Updated on 2025/04/09

写真a

 
TAKAHASHI, Yoshiyuki
 
Organization
Graduate School of Medicine Program in Integrated Medicine Medicine in Growth and Aging Professor
Graduate School
Graduate School of Medicine
Undergraduate School
School of Medicine Department of Medicine
Title
Professor

Degree 1

  1. Doctor of Medical Science ( 2000.3   Nagoya University School ofMedicine ) 

Research Interests 5

  1. Stem cell transplantation

  2. Immunology

  3. Oncology

  4. Hematology

  5. Pediatrics

Research Areas 4

  1. Others / Others  / Immunology

  2. Others / Others  / Hematology and Oncology

  3. Others / Others  / Pediatrics

  4. Life Science / Hematology and medical oncology

Research History 12

  1. Nagoya University Graduate School of Medicine   Department of Pediatrics   Professor

    2016.10

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    Country:Japan

  2. Nagoya University   Graduate School of Medicine Program in Integrated Medicine Medicine in Growth and Aging   Professor

    2016.10

  3. Associate Professor, Departmen of Developmental Pediatrics, Nagoya University Graduate School of Medicine

    2011.10

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    Country:Japan

  4. Assintant Professor, Nagoya University Graduate School of Medicine

    2010.3 - 2011.9

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    Country:Japan

  5. Assintant Professor, Nagoya University Graduate School of Medicine

    2007.4 - 2010.2

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    Country:Japan

  6. Assistant Professor, Nagoya University Graduate Scholl of Medicine

    2006.4 - 2007.3

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    Country:Japan

  7. Medical Staff Fellow, Department of Pediatrics, NagoyaUniversity Graduate School of Medicine

    2006.1 - 2006.3

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    Country:Japan

  8. Medical Staff Fellow, Department of Pediatrics, Nagoya University School of Medicine

    2000.10 - 2001.3

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    Country:Japan

  9. Medical Staff Fellow, Meijo Hospital

    2000.4 - 2000.9

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    Country:Japan

  10. Medical Staff Fellow, Department of Pediatrics, Nagoya University School of Medicine

    1995.4 - 1996.3

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    Country:Japan

  11. Medical Staff Fellow, Children's Medical Center, Japanese Red Cross Hospital

    1994.4 - 1995.3

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    Country:Japan

  12. Internship, Japanese Red Cross Nagoya First Hospital

    1992.5 - 1994.3

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    Country:Japan

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Education 2

  1. Nagoya University Graduate School of Medicine   Department of Pediatrics   Hematology and Oncology

    1996.4 - 2000.3

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    Country: Japan

  2. Nagoya University   School of Medicine   School of Medicine

    1986.4 - 1992.3

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    Country: Japan

Professional Memberships 7

  1. The Japan Society for Hematopoietic Cell Transplantation   board member

    2016.4

  2. The Japanese Society of Pediatric Hematology/Oncology

  3. The Japanese Society of Hematology

  4. Japanese Pediatrics Society

  5. Japanese Cancer Association

  6. Society of Immunotherapy for Hematological Disorders

  7. American Society of Hematology

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Committee Memberships 4

  1. Stem cell transplantaion comittee, Japan Pediatric Lukemia/Lymphoma Study Group   Stem cell transplantation comittee  

    2010.10   

  2. Solid Tumor Working Group, The Japan Society for Hematopoietic Cell   Solid tumor Working Group  

    2010.10   

  3. HLA Working Group, The Japan Society for Hematopoietic Cell   HLA Working Group  

    2010.10   

  4. Pediatric ALL Working Group, The Japan Society for Hematopoietic Cell   Solid tumor Working Group  

    2010.10   

 

Papers 517

  1. Dyskeratosis Congenita Complicated by Pulmonary Fibrosis and Myelodysplastic Syndrome with a Germline Mutation of the DKC1 Gene and a Somatic Mutation of the U2AF1 Gene in Leukocytes.

    Watanabe H, Takahashi Y, Namiki T, Nakagawa R, Inui T, Ishikawa H, Wakamatsu M, Muramatsu H, Sakamoto T

    Internal medicine (Tokyo, Japan)     2025.4

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.2169/internalmedicine.5153-24

    PubMed

  2. Early discontinuation of steroid treatment in children with abdominal pain due to IgA vasculitis.

    Kambara S, Nishio N, Sugiyama Y, Nishio Y, Takamoto Y, Kitai F, Takahashi Y, Hayashi N, Haruta K, Kondo M, Oike N, Miwa T, Watanabe N, Omori M, Kinoshita F, Furukawa T, Kawada JI, Kidokoro H, Sato Y, Takahashi Y, Nagoya Collaborative Clinical Research Team

    European journal of pediatrics   Vol. 184 ( 5 ) page: 279   2025.4

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1007/s00431-025-06107-7

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  3. Adjunctive effects of eltrombopag on immunosuppressive therapy for childhood aplastic anemia.

    Eguchi K, Ishimura M, Ohga S, Endo S, Saito S, Kamimura S, Keino D, Kato S, Azuma Y, Watanabe A, Inoue A, Higa T, Ozono S, Fujita N, Watanabe K, Takahashi Y, Japan Childhood Aplastic Anemia Study Group

    International journal of hematology   Vol. 121 ( 4 ) page: 533 - 542   2025.4

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1007/s12185-024-03903-z

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  4. Diffuse but Non-homogeneous Brain Atrophy: Identification of Specific Brain Regions and Their Correlation with Clinical Severity in Rett Syndrome

    Hajime Narita, Jun Natsume, Takeshi Suzuki, Tadashi Shiohama, Masahiro Kawaguchi, Masaki Okazaki, Atsushi Hashizume, Shinji Naganawa, Yuji Ito, Hiroyuki Yamamoto, Tomohiko Nakata, Hiroyuki Kidokoro, Yoshiyuki Takahashi, Satoru Takahashi, Keita Tsujimura

    Brain and Development   Vol. 47 ( 3 ) page: 104348 - 104348   2025.3

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    Language:English   Publishing type:Research paper (scientific journal)   Publisher:Elsevier BV  

    DOI: 10.1016/j.braindev.2025.104348

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  5. Comparable impact of lymph node metastases in T2 gallbladder cancer on postoperative prognosis irrespective of the extent of the metastases: A retrospective analysis.

    Kishi Y, Sugiura T, Mizuno T, Ito H, Takahashi Y, Noji T, Abe Y, Otsuka S, Kawakatsu S, Kato A, Tanaka M, Ebata T, Hirano S

    Journal of hepato-biliary-pancreatic sciences     2025.3

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1002/jhbp.12140

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  6. Direct Extraction and Evaluation of Intraluminal Vesicles Inside a Single Cell.

    Ida H, Yoshida T, Kumatani A, Hanayama R, Takahashi Y

    Nano letters   Vol. 25 ( 11 ) page: 4322 - 4329   2025.3

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1021/acs.nanolett.4c06315

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  7. Characteristics of neonatal-onset and presumed neonatal arterial ischemic stroke.

    Ueda K, Kataoka E, Natsume J, Hori I, Tsuji T, Miyake M, Suzuki M, Kobayashi S, Kurahashi H, Sato Y, Okumura A, Yoshikawa T, Saitoh S, Takahashi Y

    Brain & development   Vol. 47 ( 3 ) page: 104343   2025.3

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1016/j.braindev.2025.104343

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  8. Retrospective nationwide survey of pediatric RDD in Japan: a high prevalence of mutations in the kinase pathway genes.

    Asano T, Morimoto A, Nakazawa A, Ueda T, Ogawa C, Sakata N, Sugimoto K, On J, Takahashi Y, Otsuki N, Taneyama Y, Hyakuna N, Ishihara T, Matsumine A, Shioda Y, Sakamoto K, Nakazawa Y, Yasumi T, Doi T, Koga Y, Histiocytosis Study Group of the Japanese Society of Pediatric Hematology/Oncology

    International journal of hematology     2025.3

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1007/s12185-025-03962-w

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  9. Predicting Postoperative Neurological Outcomes in Metastatic Spinal Tumor Surgery Using Machine Learning.

    Maki S, Shiratani Y, Orita S, Suzuki A, Tamai K, Shimizu T, Kakutani K, Kanda Y, Tominaga H, Kawamura I, Ishihara M, Paku M, Takahashi Y, Funayama T, Miura K, Shirasawa E, Inoue H, Kimura A, Iimura T, Moridaira H, Nakajima H, Watanabe S, Akeda K, Takegami N, Nakanishi K, Sawada H, Matsumoto K, Funaba M, Suzuki H, Funao H, Oshigiri T, Hirai T, Otsuki B, Kobayakawa K, Uotani K, Manabe H, Tanishima S, Hashimoto K, Iwai C, Yamabe D, Hiyama A, Seki S, Kato K, Miyazaki M, Watanabe K, Nakamae T, Kaito T, Nakashima H, Nagoshi N, Inoue G, Imagama S, Watanabe K, Kato S, Ohtori S, Furuya T, JASA Study Group

    Spine     2025.3

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1097/BRS.0000000000005322

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  10. Plasma Proteomic Profiles of Pediatric Patients With Human Herpesvirus 6B Encephalitis Following Umbilical Cord Blood Transplantation

    Kazunori Haruta, Yuto Fukuda, Hisateru Yamaguchi, Yoshiki Kawamura, Takako Suzuki, Yuka Torii, Atsushi Narita, Hideki Muramatsu, Hiroyuki Kidokoro, Jun Natsume, Yoshiyuki Takahashi, Tetsushi Yoshikawa, Jun‐ichi Kawada

    Journal of Medical Virology   Vol. 97 ( 3 ) page: e70311   2025.3

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    Language:English   Publishing type:Research paper (scientific journal)   Publisher:Wiley  

    ABSTRACT

    Human herpesvirus 6B (HHV‐6B) encephalitis is a rare but severe complication of hematopoietic cell transplantation. This study investigated the pathogenesis of HHV‐6B encephalitis by comparing plasma proteomic profiles of four pediatric patients with HHV‐6B encephalitis to three with asymptomatic HHV‐6B reactivation following umbilical cord blood transplantation (UCBT). Plasma proteomic profiling was conducted using liquid chromatography‐mass spectrometry. Overall, 260 proteins were identified and quantified in plasma samples. At the onset of HHV‐6B encephalitis and asymptomatic reactivation, 20 and 24 proteins, respectively, were significantly upregulated compared to their respective pre‐onset levels. Of these, 11 proteins were uniquely upregulated in HHV‐6B encephalitis. S100‐A9 and S100‐A8 were the most and second‐most upregulated proteins in HHV‐6B encephalitis, respectively. Elevated plasma S100A8/A9 heterodimer levels were confirmed via enzyme‐linked immunosorbent assay in three of the four patients with HHV‐6B encephalitis. Pathway analysis identified neutrophil degranulation as the most enriched category among upregulated proteins in HHV‐6B encephalitis. Additionally, proteins related to the protein‐lipid complex remodeling pathway were more prominently upregulated in HHV‐6B encephalitis than in asymptomatic reactivation. Proteomic analysis revealed distinct plasma protein profiles between HHV‐6B encephalitis and asymptomatic HHV‐6B reactivation in pediatric UCBT recipients. The inflammatory response mediated by S100A8/A9 proteins may play a critical role in the pathogenesis of HHV‐6B encephalitis. These findings indicate that proteomic analysis may provide novel insights into the host response to HHV‐6B reactivation and the subsequent development of HHV‐6B encephalitis.

    DOI: 10.1002/jmv.70311

    PubMed

  11. Increased response to granulocyte-macrophage colony-stimulating factor in peripheral blood cells and transient manifestations mimicking juvenile myelomonocytic leukemia in a male patient with NEMO deficiency caused by a deep intronic pathogenic variant of IKBKG.

    Ueki M, Hirabayashi S, Honda Y, Takezaki S, Ohata H, Abdrabou SSMA, Sawai S, Terashita Y, Cho Y, Muramatsu H, Izawa K, Yasumi T, Takahashi Y, Yamada M, Manabe A

    Immunological medicine   Vol. 48 ( 1 ) page: 94 - 101   2025.3

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1080/25785826.2024.2422639

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  12. Successful Treatment with Sirolimus of a Patient with a Novel CARD11 Germline Mutation in B-Cell Expansion with Nuclear Factor Kappa B and T-Cell Anergy: Case Report and Literature Review.

    Nishinosono T, Muramatsu H, Wakamatsu M, Yamashita D, Fukasawa T, Shirakawa Y, Sajiki D, Maemura R, Tsumura Y, Yamamori A, Narita K, Kataoka S, Narita A, Nishio N, Miyajima Y, Takahashi Y

    Journal of clinical immunology   Vol. 45 ( 1 ) page: 78   2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1007/s10875-025-01872-4

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  13. Application of simple preparation for pediatric magnetic resonance imaging: a multicenter study in Japan.

    Sawada N, Kawada JI, Kito S, Takeo T, Narita H, Fukushima M, Masuda Y, Mitsumatsu T, Kawano Y, Sakaguchi Y, Nishio N, Kidokoro H, Sato Y, Takahashi Y, Nagoya Collaborative Clinical Research Team

    Pediatric radiology     2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1007/s00247-025-06194-6

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  14. Nanoscale visualization of the anti-tumor effect of a plasma-activated Ringer's lactate solution.

    Usuda J, Yagyu K, Tanaka H, Hori M, Ishikawa K, Takahashi Y

    Faraday discussions   Vol. 257 ( 0 ) page: 212 - 223   2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1039/d4fd00116h

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  15. Motor and functional characteristics in school-age survivors of congenital diaphragmatic hernia: a cross-sectional observational study.

    Mitsumatsu T, Ito Y, Muramatsu Y, Sato Y, Ito T, Narahara S, Miura R, Yamamoto H, Ito M, Shiraki A, Nakata T, Kotani T, Natsume J, Hayakawa M, Takahashi Y, Kidokoro H

    Archives of disease in childhood. Fetal and neonatal edition     2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1136/archdischild-2024-327942

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  16. Surgical strategy for metastatic spinal tumors based on Spine Instability Neoplastic Score and patient-reported outcomes: JASA multicenter prospective study.

    Nakajima H, Watanabe S, Honjoh K, Kubota A, Shiratani Y, Suzuki A, Terai H, Shimizu T, Kakutani K, Kanda Y, Tominaga H, Kawamura I, Ishihara M, Paku M, Takahashi Y, Funayama T, Miura K, Shirasawa E, Inoue H, Kimura A, Iimura T, Moridaira H, Akeda K, Takegami N, Nakanishi K, Sawada H, Matsumoto K, Funaba M, Suzuki H, Funao H, Oshigiri T, Hirai T, Otsuki B, Kobayakawa K, Uotani K, Manabe H, Tanishima S, Hashimoto K, Iwai C, Yamabe D, Hiyama A, Seki S, Goto Y, Miyazaki M, Watanabe K, Nakamae T, Kaito T, Nakashima H, Nagoshi N, Kato S, Imagama S, Watanabe K, Inoue G, Furuya T

    Journal of neurosurgery. Spine   Vol. 42 ( 2 ) page: 203 - 214   2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.3171/2024.7.SPINE24340

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  17. Real-world Outcomes of Commercial Tisagenlecleucel for Children, Adolescents, and Young Adults With Acute Lymphoblastic Leukemia in Japan.

    Kato I, Tomizawa D, Kato M, Hirabayashi S, Manabe A, Irie M, Sasahara Y, Arakawa Y, Koh K, Sakaguchi H, Sugiyama M, Ogawa C, Kamiya T, Saito S, Nakazawa Y, Nishio N, Takahashi Y, Iwai N, Adachi S, Takita J, Miyamura T, Yokoyama S, Oba U, Ueda T, Koga Y, Hiramatsu H

    Transplantation and cellular therapy   Vol. 31 ( 2 ) page: 86 - 96   2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1016/j.jtct.2024.11.016

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  18. Mechanical signaling through membrane tension induces somal translocation during neuronal migration.

    Minegishi T, Hasebe H, Aoyama T, Naruse K, Takahashi Y, Inagaki N

    The EMBO journal   Vol. 44 ( 3 ) page: 767 - 780   2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1038/s44318-024-00326-8

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  19. Cord Blood Transplantation Using Myeloablative Conditioning for Pediatric Advanced Myelodysplastic Syndrome in AMeD Syndrome With a Novel ADH5 Variant.

    Morishita Y, Hamada M, Uemura S, Kikuchi H, Hisakawa H, Fujikawa T, Tamura A, Okuno Y, Wakamatsu M, Muramatsu H, Takahashi Y, Hasegawa D, Kosaka Y

    Pediatric blood & cancer   Vol. 72 ( 2 ) page: e31465   2025.2

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1002/pbc.31465

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  20. Identification of a Novel Stomatal Opening Chemical, PP242, That Inhibits Early Abscisic Acid Signal Transduction in Guard Cells.

    Oh A, Kimura R, Inoue S, Sato T, Hayashi Y, Sato A, Takahashi Y, Kinoshita T

    Plant & cell physiology     2025.1

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1093/pcp/pcaf013

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  21. EMT-Induced Morphological Variations on Living Cell Membrane Surface.

    Ida H, Taira N, Nashimoto Y, Kumatani A, Takahashi Y, Shiku H

    Analytical chemistry   Vol. 97 ( 1 ) page: 312 - 318   2025.1

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1021/acs.analchem.4c04204

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  22. Lattice-guided growth of dense arrays of aligned transition metal dichalcogenide nanoribbons with high catalytic reactivity.

    Ma Z, Solís-Fernández P, Hirata K, Lin YC, Shinokita K, Maruyama M, Honda K, Kato T, Uchida A, Ogura H, Otsuka T, Hara M, Matsuda K, Suenaga K, Okada S, Kato T, Takahashi Y, Ago H

    Science advances   Vol. 11 ( 2 ) page: eadr8046   2025.1

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1126/sciadv.adr8046

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  23. Association between adeno-associated virus 2 and severe acute hepatitis of unknown etiology in Japanese children Reviewed

    Ken-ichi Iwata, Yuka Torii, Aiko Sakai, Yuto Fukuda, Kazunori Haruta, Makoto Yamaguchi, Takako Suzuki, Yuri Etani, Yoshiyuki Takahashi, Shuichiro Umetsu, Ayano Inui, Ryo Sumazaki, Jun-ichi Kawada

    Journal of Infection and Chemotherapy   Vol. 31 ( 1 ) page: 102462   2025.1

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    Language:English   Publishing type:Research paper (scientific journal)   Publisher:Elsevier BV  

    DOI: 10.1016/j.jiac.2024.07.002

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  24. Corrosion Inspection for Hard Disk Media with Carbon-Based Overcoats by In-Liquid Open-Loop Electric Potential Microscopy.

    Hirata K, Omi JI, Taniguchi D, Miyazawa K, Komatsu F, Takahashi Y, Fukuma T

    ACS applied materials & interfaces   Vol. 16 ( 50 ) page: 70020 - 70027   2024.12

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1021/acsami.4c14138

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  25. Sleep state-dependent development of resting-state functional connectivity during the preterm period.

    Shiraki A, Kidokoro H, Watanabe H, Taga G, Ushida T, Narita H, Mitsumatsu T, Kumai S, Suzui R, Sawamura F, Ito Y, Yamamoto H, Nakata T, Sato Y, Hayakawa M, Takahashi Y, Natsume J

    Sleep   Vol. 47 ( 12 )   2024.12

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1093/sleep/zsae225

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  26. Prospective Registration Study for Establishing Minimal Clinically Important Differences in Patients Undergoing Surgery for Spinal Metastases.

    Hirota R, Oshigiri T, Iesato N, Emori M, Teramoto A, Shiratani Y, Suzuki A, Terai H, Shimizu T, Kakutani K, Kanda Y, Tominaga H, Kawamura I, Ishihara M, Paku M, Takahashi Y, Funayama T, Miura K, Shirasawa E, Inoue H, Kimura A, Iimura T, Moridaira H, Nakajima H, Watanabe S, Akeda K, Takegami N, Nakanishi K, Sawada H, Matsumoto K, Funaba M, Suzuki H, Funao H, Hirai T, Otsuki B, Kobayakawa K, Uotani K, Manabe H, Tanishima S, Hashimoto K, Iwai C, Yamabe D, Hiyama A, Seki S, Goto Y, Miyazaki M, Watanabe K, Nakamae T, Kaito T, Nakashima H, Nagoshi N, Kato S, Imagama S, Watanabe K, Inoue G, Furuya T

    Spine   Vol. 49 ( 22 ) page: 1539 - 1547   2024.11

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1097/BRS.0000000000005062

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  27. Ruxolitinib for pediatric patients with treatment-naïve and steroid-refractory acute graft-versus-host disease: the REACH4 study.

    Locatelli F, Kang HJ, Bruno B, Gandemer V, Rialland F, Faraci M, Takahashi Y, Koh K, Bittencourt H, Cleary G, Rosko C, Li X, St Pierre A, Prahallad A, Diaz-de-Heredia C

    Blood   Vol. 144 ( 20 ) page: 2095 - 2106   2024.11

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1182/blood.2023022565

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  28. Effects of Palliative Surgical Treatment for Spinal Metastases on the Patient's Quality of Life With a Focus on the Segment of the Metastasis: A Prospective Multicenter Study.

    Segi N, Nakashima H, Ito S, Ouchida J, Shiratani Y, Shimizu T, Suzuki A, Terai H, Kakutani K, Kanda Y, Tominaga H, Kawamura I, Ishihara M, Paku M, Takahashi Y, Funaba M, Funayama T, Nakajima H, Akeda K, Hirai T, Inoue H, Nakanishi K, Funao H, Oshigiri T, Otsuki B, Kobayakawa K, Tanishima S, Hashimoto K, Iimura T, Sawada H, Uotani K, Manabe H, Iwai C, Yamabe D, Hiyama A, Seki S, Goto Y, Miyazaki M, Watanabe K, Nakamae T, Kaito T, Nagoshi N, Kato S, Watanabe K, Imagama S, Inoue G, Furuya T

    Global spine journal     page: 21925682241297948   2024.11

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1177/21925682241297948

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  29. Frequent loss of HLA-B*40:02 from leucocytes in male patients with hepatitis-associated aplastic anaemia.

    Katagiri T, Takahashi R, Zaimoku Y, Hosokawa K, Ogawa S, Takahashi Y, Miyamura T, Onishi Y, Koike T, Nishida T, Uchida N, Doki N, Tanaka M, Eto T, Itagaki M, Sawa M, Fukuda T, Onizuka M, Atsuta Y, Nakao S

    British journal of haematology   Vol. 205 ( 5 ) page: 2084 - 2088   2024.11

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1111/bjh.19808

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  30. Association between combined use of epidural analgesia and oxytocin administration during labor and offspring outcomes: a narrative review and proposal.

    Tachi A, Takahashi Y, Kotani T

    Nagoya journal of medical science   Vol. 86 ( 4 ) page: 549 - 563   2024.11

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.18999/nagjms.86.4.549

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  31. A Retrospective Study of Pediatric Patients With Low- or Intermediate-Risk Acute Myeloid Leukemia Who Underwent Allogeneic Hematopoietic Cell Transplantation for the AML-05 Study Conducted by the Japanese Pediatric Leukemia/Lymphoma Study Group.

    Hashii Y, Kawaguchi K, Kurakami H, Umeda K, Hasegawa D, Taki T, Hyakuna N, Ishida H, Takahashi Y, Nagasawa M, Yabe H, Yano M, Nakazawa Y, Fujisaki H, Matsumoto K, Yanagimachi M, Yoshida N, Kakuda H, Satou A, Tabuchi K, Tomizawa D, Taga T, Adachi S, Koh K, Kato K

    Transplantation and cellular therapy   Vol. 30 ( 11 ) page: 1102.e1 - 1102.e12   2024.11

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1016/j.jtct.2024.08.011

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  32. The efficacy of wrapping with polyglycolic acid mesh and fibrin glue in preventing clinically relevant pancreatic fistula after minimally invasive distal pancreatectomy (WRAP Study): study protocol for a multicenter randomized controlled trial in Japan.

    Baba H, Oba A, Tanaka K, Miura T, Ban D, Edanami M, Ishikawa Y, Ohgi K, Tanaka H, Shintakuya R, Ikenaga N, Ijichi T, Kiya Y, Muranushi R, Yamaki S, Miyazaki N, Takeuchi S, Aoki S, Mizui T, Tanaka M, Ueda H, Dei H, Takami H, Okada K, Nakata K, Mataki Y, Osakabe H, Shibuya K, Hashimoto D, Inoue Y, Hirano S, Unno M, Esaki M, Kitago M, Akahoshi K, Sugiura T, Ebata T, Uemura K, Nakamura M, Otsuka T, Nagakawa Y, Fujii T, Satoi S, Takahashi Y

    BMC surgery   Vol. 24 ( 1 ) page: 314   2024.10

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1186/s12893-024-02610-0

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  33. Statistically and functionally fine-mapped blood eQTLs and pQTLs from 1,405 humans reveal distinct regulation patterns and disease relevance.

    Wang QS, Hasegawa T, Namkoong H, Saiki R, Edahiro R, Sonehara K, Tanaka H, Azekawa S, Chubachi S, Takahashi Y, Sakaue S, Namba S, Yamamoto K, Shiraishi Y, Chiba K, Tanaka H, Makishima H, Nannya Y, Zhang Z, Tsujikawa R, Koike R, Takano T, Ishii M, Kimura A, Inoue F, Kanai T, Fukunaga K, Ogawa S, Imoto S, Miyano S, Okada Y, Japan COVID-19 Task Force

    Nature genetics   Vol. 56 ( 10 ) page: 2054 - 2067   2024.10

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    Language:English   Publishing type:Research paper (scientific journal)  

    DOI: 10.1038/s41588-024-01896-3

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  34. Publisher Correction: Statistically and functionally fine-mapped blood eQTLs and pQTLs from 1,405 humans reveal distinct regulation patterns and disease relevance.

    Wang QS, Hasegawa T, Namkoong H, Saiki R, Edahiro R, Sonehara K, Tanaka H, Azekawa S, Chubachi S, Takahashi Y, Sakaue S, Namba S, Yamamoto K, Shiraishi Y, Chiba K, Tanaka H, Makishima H, Nannya Y, Zhang Z, Tsujikawa R, Koike R, Takano T, Ishii M, Kimura A, Inoue F, Kanai T, Fukunaga K, Ogawa S, Imoto S, Miyano S, Okada Y, Japan COVID-19 Task Force

    Nature genetics   Vol. 56 ( 10 ) page: 2281   2024.10

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  49. Combination of Measurements of the Top Quark Mass from Data Collected by the ATLAS and CMS Experiments at sqrt[s]=7 and 8 TeV.

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  56. Integrated proteogenomic analysis for inherited bone marrow failure syndrome. International journal

    Manabu Wakamatsu, Hideki Muramatsu, Hironori Sato, Masaki Ishikawa, Ryo Konno, Daisuke Nakajima, Motoharu Hamada, Yusuke Okuno, Yusuke Kawashima, Asahito Hama, Masafumi Ito, Hideto Iwafuchi, Yoshiyuki Takahashi, Osamu Ohara

    Leukemia   Vol. 38 ( 6 ) page: 1256 - 1265   2024.6

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    Recent advances in in-depth data-independent acquisition proteomic analysis have enabled comprehensive quantitative analysis of >10,000 proteins. Herein, an integrated proteogenomic analysis for inherited bone marrow failure syndrome (IBMFS) was performed to reveal their biological features and to develop a proteomic-based diagnostic assay in the discovery cohort; dyskeratosis congenita (n = 12), Fanconi anemia (n = 11), Diamond-Blackfan anemia (DBA, n = 9), Shwachman-Diamond syndrome (SDS, n = 6), ADH5/ALDH2 deficiency (n = 4), and other IBMFS (n = 18). Unsupervised proteomic clustering identified eight independent clusters (C1-C8), with the ribosomal pathway specifically downregulated in C1 and C2, enriched for DBA and SDS, respectively. Six patients with SDS had significantly decreased SBDS protein expression, with two of these not diagnosed by DNA sequencing alone. Four patients with ADH5/ALDH2 deficiency showed significantly reduced ADH5 protein expression. To perform a large-scale rapid IBMFS screening, targeted proteomic analysis was performed on 417 samples from patients with IBMFS-related hematological disorders (n = 390) and healthy controls (n = 27). SBDS and ADH5 protein expressions were significantly reduced in SDS and ADH5/ALDH2 deficiency, respectively. The clinical application of this first integrated proteogenomic analysis would be useful for the diagnosis and screening of IBMFS, where appropriate clinical screening tests are lacking.

    DOI: 10.1038/s41375-024-02263-1

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  57. Determinants of postpartum depression among women in Kampong Chhnang Province, Cambodia.

    Guechhorng I, Inthaphatha S, Nishino K, Takahashi Y, Hamajima N, Yamamoto E

    Archives of psychiatric nursing   Vol. 50   page: 60 - 66   2024.6

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    DOI: 10.1016/j.apnu.2024.03.007

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  58. Novel air-liquid interface culture model to investigate stiffness-dependent behaviors of alveolar epithelial cells.

    Takahashi Y, Ito S, Wang J, Kim J, Matsumoto T, Maeda E

    Biochemical and biophysical research communications   Vol. 708   page: 149791   2024.5

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    DOI: 10.1016/j.bbrc.2024.149791

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  59. Scoring system for diagnosis and pretreatment risk assessment of neuroblastoma using urinary biomarker combinations. International journal

    Hizuru Amano, Hiroo Uchida, Kazuharu Harada, Atsushi Narita, Shigehisa Fumino, Yuji Yamada, Shun Kumano, Mayumi Abe, Takashi Ishigaki, Minoru Sakairi, Chiyoe Shirota, Takahisa Tainaka, Wataru Sumida, Kazuki Yokota, Satoshi Makita, Shuhei Karakawa, Yuichi Mitani, Shojiro Matsumoto, Yutaka Tomioka, Hideki Muramatsu, Nobuhiro Nishio, Tsuyoshi Osawa, Masataka Taguri, Katsuyoshi Koh, Tatsuro Tajiri, Motohiro Kato, Kimikazu Matsumoto, Yoshiyuki Takahashi, Akinari Hinoki

    Cancer science   Vol. 115 ( 5 ) page: 1634 - 1645   2024.5

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    The urinary catecholamine metabolites, homovanillic acid (HVA) and vanillylmandelic acid (VMA), are used for the adjunctive diagnosis of neuroblastomas. We aimed to develop a scoring system for the diagnosis and pretreatment risk assessment of neuroblastoma, incorporating age and other urinary catecholamine metabolite combinations. Urine samples from 227 controls (227 samples) and 68 patients with neuroblastoma (228 samples) were evaluated. First, the catecholamine metabolites vanillactic acid (VLA) and 3-methoxytyramine sulfate (MTS) were identified as urinary marker candidates through comprehensive analysis using liquid chromatography-mass spectrometry. The concentrations of these marker candidates and conventional markers were then compared among controls, patients, and numerous risk groups to develop a scoring system. Participants were classified into four groups: control, low risk, intermediate risk, and high risk, and the proportional odds model was fitted using the L2-penalized maximum likelihood method, incorporating age on a monthly scale for adjustment. This scoring model using the novel urine catecholamine metabolite combinations, VLA and MTS, had greater area under the curve values than the model using HVA and VMA for diagnosis (0.978 vs. 0.964), pretreatment risk assessment (low and intermediate risk vs. high risk: 0.866 vs. 0.724; low risk vs. intermediate and high risk: 0.871 vs. 0.680), and prognostic factors (MYCN status: 0.741 vs. 0.369, histology: 0.932 vs. 0.747). The new system also had greater accuracy in detecting missing high-risk neuroblastomas, and in predicting the pretreatment risk at the time of screening. The new scoring system employing VLA and MTS has the potential to replace the conventional adjunctive diagnostic method using HVA and VMA.

    DOI: 10.1111/cas.16116

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  60. Effect of levodopa on pathological gait in Dravet syndrome: A randomized crossover trial using three-dimensional gait analysis.

    Suzuki T, Natsume J, Ito Y, Ito T, Noritake K, Kinoshita F, Fukasawa T, Tsuji T, Itomi K, Kurahashi H, Kubota K, Okanishi T, Saitoh S, Sugiura H, Watanabe H, Takahashi Y, Kidokoro H

    Epilepsia   Vol. 65 ( 5 ) page: 1304 - 1313   2024.5

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    DOI: 10.1111/epi.17888

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  61. Comparison of Whole Blood and Plasma for Monitoring Cytomegalovirus and Epstein-Barr Virus.

    Fukuda Y, Torii Y, Iwata KI, Haruta K, Yamaguchi M, Suzuki T, Narita A, Muramatsu H, Ogura Y, Takahashi Y, Ito Y, Kawada JI

    The new microbiologica   Vol. 47 ( 1 ) page: 52 - 59   2024.5

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  62. Single-Cell Transcriptomic Analysis of Epstein-Barr Virus-Associated Hemophagocytic Lymphohistiocytosis

    Takako Suzuki, Yoshitaka Sato, Yusuke Okuno, Yuka Torii, Yuto Fukuda, Kazunori Haruta, Makoto Yamaguchi, Yoshiki Kawamura, Asahito Hama, Atsushi Narita, Hideki Muramatsu, Tetsushi Yoshikawa, Yoshiyuki Takahashi, Hiroshi Kimura, Yoshinori Ito, Jun-ichi Kawada

    Journal of Clinical Immunology   Vol. 44 ( 4 ) page: 103   2024.4

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    Language:English   Publishing type:Research paper (scientific journal)   Publisher:Springer Science and Business Media LLC  

    DOI: 10.1007/s10875-024-01701-0

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    Other Link: https://link.springer.com/article/10.1007/s10875-024-01701-0/fulltext.html

  63. Nanopore sequencing in distinguishing between wild-type and vaccine strains of Varicella-Zoster virus. International journal

    Yuto Fukuda, Takako Suzuki, Ken-Ichi Iwata, Kazunori Haruta, Makoto Yamaguchi, Yuka Torii, Atsushi Narita, Hideki Muramatsu, Yoshiyuki Takahashi, Jun-Ichi Kawada

    Vaccine   Vol. 42 ( 11 ) page: 2927 - 2932   2024.4

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    BACKGROUND: The introduction of varicella vaccines into routine pediatric immunization programs has led to a considerable reduction in varicella incidence. However, there have been reports of varicella, herpes zoster, and meningitis caused by the vaccine strain of varicella-zoster virus (VZV), raising concerns. Establishing the relationship between the wild-type and vaccine strains in VZV infections among previously vaccinated individuals is crucial. Differences in the single nucleotide polymorphisms (SNPs) among vaccine strains can be utilized to identify the strain. In this study, we employed nanopore sequencing to identify VZV strains and analyzed clinical samples. METHODS: We retrospectively examined vesicle and cerebrospinal fluid samples from patients with VZV infections. One sample each of the wild-type and vaccine strains, previously identified using allelic discrimination real-time PCR and direct sequencing, served as controls. Ten samples with undetermined VZV strains were included. After DNA extraction, a long PCR targeting the VZV ORF62 region was executed. Nanopore sequencing identified SNPs, allowing discrimination between the vaccine and wild-type strains. RESULTS: Nanopore sequencing confirmed SNPs at previously reported sites (105,705, 106,262, 107,136, and 107,252), aiding in distinguishing between wild-type and vaccine strains. Among the ten unknown samples, nine were characterized as wild strains and one as a vaccine strain. Even in samples with low VZV DNA levels, nanopore sequencing was effective in strain identification. CONCLUSION: This study validates that nanopore sequencing is a reliable method for differentiating between the wild-type and vaccine strains of VZV. Its ability to produce long-read sequences is remarkable, allowing simultaneous confirmation of known SNPs and the detection of new mutations. Nanopore sequencing can serve as a valuable tool for the swift and precise identification of wild-type and vaccine strains and has potential applications in future VZV surveillance.

    DOI: 10.1016/j.vaccine.2024.03.046

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  64. Nationwide survey of patients with multisystem proteinopathy in Japan.

    Yamashita S, Takahashi Y, Hashimoto J, Murakami A, Nakamura R, Katsuno M, Izumi R, Suzuki N, Warita H, Aoki M, Japan MSP Study Group

    Annals of clinical and translational neurology   Vol. 11 ( 4 ) page: 938 - 945   2024.4

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    DOI: 10.1002/acn3.52011

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  65. Multi-color polymer carbon dots synthesized from waste polyolefins through phenylenediamine-assisted hydrothermal processing.

    Takahashi Y, Chan K, Zinchenko A

    Chemosphere   Vol. 354   page: 141685   2024.4

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    DOI: 10.1016/j.chemosphere.2024.141685

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  66. Comparison of the Outcomes of Plating, Screw Fixation, and Pinning in Sanders Type II Fractures: A Multicenter (TRON) Retrospective Study.

    Kato M, Takegami Y, Tokutake K, Asami Y, Takahashi Y, Takahashi H, Kumagai H, Imagama S

    The Journal of foot and ankle surgery : official publication of the American College of Foot and Ankle Surgeons   Vol. 63 ( 2 ) page: 171 - 175   2024.3

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    DOI: 10.1053/j.jfas.2023.10.002

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  67. Most axonal mitochondria in cortical pyramidal neurons lack mitochondrial DNA and consume ATP.

    Hirabayashi Y, Lewis TL Jr, Du Y, Virga DM, Decker AM, Coceano G, Alvelid J, Paul MA, Hamilton S, Kneis P, Takahashi Y, Gaublomme JT, Testa I, Polleux F

    bioRxiv : the preprint server for biology     2024.2

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    DOI: 10.1101/2024.02.12.579972

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  68. IκB kinase phosphorylates cytoplasmic TDP-43 and promotes its proteasome degradation.

    Iguchi Y, Takahashi Y, Li J, Araki K, Amakusa Y, Kawakami Y, Kobayashi K, Yokoi S, Katsuno M

    The Journal of cell biology   Vol. 223 ( 2 )   2024.2

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    DOI: 10.1083/jcb.202302048

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  69. Dysbiosis of gut microbiota in patients with protein-losing enteropathy after the Fontan procedure.

    Go K, Horiba K, Yamamoto H, Morimoto Y, Fukasawa Y, Ohashi N, Yasuda K, Ishikawa Y, Kuraishi K, Suzuki K, Ito Y, Takahashi Y, Kato T

    International journal of cardiology   Vol. 396   page: 131554   2024.2

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    DOI: 10.1016/j.ijcard.2023.131554

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  70. 愛知県における副腎白質ジストロフィー新生児マススクリーニングの2年間の実績と課題

    中田 智彦, 伊藤 祐史, 若松 学, 村松 秀城, 山本 啓之, 城所 博之, 久保田 一生, 山田 晋一郎, 勝野 雅央, 伊藤 哲哉, 下澤 伸行, 高橋 義行, 夏目 淳

    日本小児科学会雑誌   Vol. 128 ( 2 ) page: 283 - 283   2024.2

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  71. Factors associated with perineal pain on the first postnatal day after vaginal delivery: a cross-sectional study of primiparous women.

    Yamada A, Takahashi Y, Hirose M, Usami Y, Maruya S, Tamakoshi K

    Nagoya journal of medical science   Vol. 86 ( 1 ) page: 52 - 63   2024.2

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    DOI: 10.18999/nagjms.86.1.52

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  72. Comparison of plasma proteomic profiles of patients with Epstein-Barr virus-associated hemophagocytic lymphohistiocytosis and infectious mononucleosis. International journal

    Kazunori Haruta, Takako Suzuki, Makoto Yamaguchi, Yuto Fukuda, Yuka Torii, Yoshiyuki Takahashi, Yoshinori Ito, Jun-Ichi Kawada

    Journal of medical virology   Vol. 96 ( 2 ) page: e29450   2024.2

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    Primary Epstein-Barr virus (EBV) infection occasionally causes EBV-infectious mononucleosis (EBV-IM) and EBV-hemophagocytic lymphohistiocytosis (EBV-HLH). Although EBV-IM is mostly mild and self-limiting, EBV-HLH is a life-threatening disease characterized by excessive immune activation. However, the pathogenesis of EBV-HLH is yet to be fully elucidated. A diagnostic biomarker for EBV-HLH is desirable because early diagnosis and treatment are critical for the effective management of patients. In this study, the proteomic profiling of plasma was performed using liquid chromatography-mass spectrometry to identify proteins specific to EBV-IM and EBV-HLH. Furthermore, pathway analysis was performed for the proteins upregulated in patients with EBV-IM and EBV-HLH. Compared to healthy controls, 63 and 18 proteins were upregulated in patients with EBV-IM and EBV-HLH, respectively. Pathway and process enrichment analyses revealed that the complement system was the most enriched category of upregulated proteins in EBV-IM, whereas proteins related to immune effector processes were the most enriched in EBV-HLH. Among the 18 proteins upregulated in EBV-HLH, seven were exclusive to EBV-HLH. These specific proteins were associated with three pathways, and apolipoprotein E was commonly found in all the pathways. Proteomic analysis may provide new insights into the host response to EBV infection and the pathogenesis of EBV-related diseases.

    DOI: 10.1002/jmv.29450

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  73. Nanoscrolls of Janus Monolayer Transition Metal Dichalcogenides.

    Kaneda M, Zhang W, Liu Z, Gao Y, Maruyama M, Nakanishi Y, Nakajo H, Aoki S, Honda K, Ogawa T, Hashimoto K, Endo T, Aso K, Chen T, Oshima Y, Yamada-Takamura Y, Takahashi Y, Okada S, Kato T, Miyata Y

    ACS nano   Vol. 18 ( 4 ) page: 2772 - 2781   2024.1

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    DOI: 10.1021/acsnano.3c05681

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  74. Retinoid Therapy for Neuroblastoma: Historical Overview, Regulatory Challenges, and Prospects.

    Makimoto A, Fujisaki H, Matsumoto K, Takahashi Y, Cho Y, Morikawa Y, Yuza Y, Tajiri T, Iehara T

    Cancers   Vol. 16 ( 3 )   2024.1

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    DOI: 10.3390/cancers16030544

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  75. Evidence for the Higgs Boson Decay to a Z Boson and a Photon at the LHC.

    Aad G, Abbott B, Abeling K, Abicht NJ, Abidi SH, Aboulhorma A, Abramowicz H, Abreu H, Abulaiti Y, Acharya BS, Adam Bourdarios C, Adamczyk L, Adamek L, Addepalli SV, Addison MJ, Adelman J, Adiguzel A, Adye T, Affolder AA, Afik Y, Agaras MN, Agarwala J, Aggarwal A, Agheorghiesei C, Ahmad A, Ahmadov F, Ahmed WS, Ahuja S, Ai X, Aielli G, Aikot A, Ait Tamlihat M, Aitbenchikh B, Aizenberg I, Akbiyik M, Åkesson TPA, Akimov AV, Akiyama D, Akolkar NN, Al Khoury K, Alberghi GL, Albert J, Albicocco P, Albouy GL, Alderweireldt S, Aleksa M, Aleksandrov IN, Alexa C, Alexopoulos T, Alfonsi F, Algren M, Alhroob M, Ali B, Ali HMJ, Ali S, Alibocus SW, Aliev M, Alimonti G, Alkakhi W, Allaire C, Allbrooke BMM, Allen JF, Allendes Flores CA, Allport PP, Aloisio A, Alonso F, Alpigiani C, Alvarez Estevez M, Alvarez Fernandez A, Alves Cardoso M, Alviggi MG, Aly M, Amaral Coutinho Y, Ambler A, Amelung C, Amerl M, Ames CG, Amidei D, Amor Dos Santos SP, Amos KR, Ananiev V, Anastopoulos C, Andeen T, Anders JK, Andrean SY, Andreazza A, Angelidakis S, Angerami A, Anisenkov AV, Annovi A, Antel C, Anthony MT, Antipov E, Antonelli M, Anulli F, Aoki M, Aoki T, Aparisi Pozo JA, Aparo MA, Aperio Bella L, Appelt C, Apyan A, Aranzabal N, Arcangeletti C, Arce ATH, Arena E, Arguin JF, Argyropoulos S, Arling JH, Arnaez O, Arnold H, Artoni G, Asada H, Asai K, Asai S, Asbah NA, Assahsah J, Assamagan K, Astalos R, Atashi S, Atkin RJ, Atkinson M, Atmani H, Atmasiddha PA, Augsten K, Auricchio S, Auriol AD, Austrup VA, Avolio G, Axiotis K, Azuelos G, Babal D, Bachacou H, Bachas K, Bachiu A, Backman F, Badea A, Bagnaia P, Bahmani M, Bailey AJ, Bailey VR, Baines JT, Baines L, Bakalis C, Baker OK, Bakos E, Bakshi Gupta D, Balakrishnan V, Balasubramanian R, Baldin EM, Balek P, Ballabene E, Balli F, Baltes LM, Balunas WK, Balz J, Banas E, Bandieramonte M, Bandyopadhyay A, Bansal S, Barak L, Barakat M, Barberio EL, Barberis D, Barbero M, Barel MZ, Barends KN, Barillari T, Barisits MS, Barklow T, Baron P, Baron Moreno DA, Baroncelli A, Barone G, Barr AJ, Barr JD, Barranco Navarro L, Barreiro F, Barreiro Guimarães da Costa J, Barron U, Barros Teixeira MG, Barsov S, Bartels F, Bartoldus R, Barton AE, Bartos P, Basan A, Baselga M, Bassalat A, Basso MJ, Basson CR, Bates RL, Batlamous S, Batley JR, Batool B, Battaglia M, Battulga D, Bauce M, Bauer M, Bauer P, Bazzano Hurrell LT, Beacham JB, Beau T, Beauchemin PH, Becherer F, Bechtle P, Beck HP, Becker K, Beddall AJ, Bednyakov VA, Bee CP, Beemster LJ, Beermann TA, Begalli M, Begel M, Behera A, Behr JK, Beirer JF, Beisiegel F, Belfkir M, Bella G, Bellagamba L, Bellerive A, Bellos P, Beloborodov K, Benchekroun D, Bendebba F, Benhammou Y, Benoit M, Bensinger JR, Bentvelsen S, Beresford L, Beretta M, Bergeaas Kuutmann E, Berger N, Bergmann B, Beringer J, Bernardi G, Bernius C, Bernlochner FU, Bernon F, Berry T, Berta P, Berthold A, Bertram IA, Bethke S, Betti A, Bevan AJ, Bhalla NK, Bhamjee M, Bhatta S, Bhattacharya DS, Bhattarai P, Bhopatkar VS, Bi R, Bianchi RM, Bianco G, Biebel O, Bielski R, Biglietti M, Bindi M, Bingul A, Bini C, Biondini A, Birch-Sykes CJ, Bird GA, Birman M, Biros M, Biryukov S, Bisanz T, Bisceglie E, Biswal JP, Biswas D, Bitadze A, Bjørke K, Bloch I, Blocker C, Blue A, Blumenschein U, Blumenthal J, Bobbink GJ, Bobrovnikov VS, Boehler M, Boehm B, Bogavac D, Bogdanchikov AG, Bohm C, Boisvert V, Bokan P, Bold T, Bomben M, Bona M, Boonekamp M, Booth CD, Borbély AG, Bordulev IS, Borecka-Bielska HM, Borissov G, Bortoletto D, Boscherini D, Bosman M, Bossio Sola JD, Bouaouda K, Bouchhar N, Boudreau J, Bouhova-Thacker EV, Boumediene D, Bouquet R, Boveia A, Boyd J, Boye D, Boyko IR, Bracinik J, Brahimi N, Brandt G, Brandt O, Braren F, Brau B, Brau JE, Brener R, Brenner L, Brenner R, Bressler S, Britton D, Britzger D, Brock I, Brooijmans G, Brooks WK, Brost E, Brown LM, Bruce LE, Bruckler TL, Bruckman de Renstrom PA, Brüers B, Bruni A, Bruni G, Bruschi M, Bruscino N, Buanes T, Buat Q, Buchin D, Buckley AG, Bulekov O, Bullard BA, Burdin S, Burgard CD, Burger AM, Burghgrave B, Burlayenko O, Burr JTP, Burton CD, Burzynski JC, Busch EL, Büscher V, Bussey PJ, Butler JM, Buttar CM, Butterworth JM, Buttinger W, Buxo Vazquez CJ, Buzykaev AR, Cabrera Urbán S, Cadamuro L, Caforio D, Cai H, Cai Y, Cairo VMM, Cakir O, Calace N, Calafiura P, Calderini G, Calfayan P, Callea G, Caloba LP, Calvet D, Calvet S, Calvet TP, Calvetti M, Camacho Toro R, Camarda S, Camarero Munoz D, Camarri P, Camerlingo MT, Cameron D, Camincher C, Campanelli M, Camplani A, Canale V, Canesse A, Cantero J, Cao Y, Capocasa F, Capua M, Carbone A, Cardarelli R, Cardenas JCJ, Cardillo F, Carli T, Carlino G, Carlotto JI, Carlson BT, Carlson EM, Carminati L, Carnelli A, Carnesale M, Caron S, Carquin E, Carrá S, Carratta G, Carrio Argos F, Carter JWS, Carter TM, Casado MP, Caspar M, Castiglia EG, Castillo FL, Castillo Garcia L, Castillo Gimenez V, Castro NF, Catinaccio A, Catmore JR, Cavaliere V, Cavalli N, Cavasinni V, Cekmecelioglu YC, Celebi E, Celli F, Centonze MS, Cepaitis V, Cerny K, Cerqueira AS, Cerri A, Cerrito L, Cerutti F, Cervato B, Cervelli A, Cesarini G, Cetin SA, Chadi Z, Chakraborty D, Chan J, Chan WY, Chapman JD, Chapon E, Chargeishvili B, Charlton DG, Charman TP, Chatterjee M, Chauhan C, Chekanov S, Chekulaev SV, Chelkov GA, Chen A, Chen B, Chen B, Chen H, Chen H, Chen J, Chen J, Chen M, Chen S, Chen SJ, Chen X, Chen X, Chen Y, Cheng CL, Cheng HC, Cheong S, Cheplakov A, Cheremushkina E, Cherepanova E, Cherkaoui El Moursli R, Cheu E, Cheung K, Chevalier L, Chiarella V, Chiarelli G, Chiedde N, Chiodini G, Chisholm AS, Chitan A, Chitishvili M, Chizhov MV, Choi K, Chomont AR, Chou Y, Chow EYS, Chowdhury T, Chu KL, Chu MC, Chu X, Chudoba J, Chwastowski JJ, Cieri D, Ciesla KM, Cindro V, Ciocio A, Cirotto F, Citron ZH, Citterio M, Ciubotaru DA, Ciungu BM, Clark A, Clark PJ, Clavijo Columbie JM, Clawson SE, Clement C, Clercx J, Clissa L, Coadou Y, Cobal M, Coccaro A, Barrue RFC, Coelho Lopes De Sa R, Coelli S, Cohen H, Coimbra AEC, Cole B, Collot J, Conde Muiño P, Connell MP, Connell SH, Connelly IA, Conroy EI, Conventi F, Cooke HG, Cooper-Sarkar AM, Cordeiro Oudot Choi A, Cormier F, Corpe LD, Corradi M, Corriveau F, Cortes-Gonzalez A, Costa MJ, Costanza F, Costanzo D, Cote BM, Cowan G, Cranmer K, Cremonini D, Crépé-Renaudin S, Crescioli F, Cristinziani M, Cristoforetti M, Croft V, Crosby JE, Crosetti G, Cueto A, Cuhadar Donszelmann T, Cui H, Cui Z, Cunningham WR, Curcio F, Czodrowski P, Czurylo MM, De Sousa MJDCS, Da Fonseca Pinto JV, Da Via C, Dabrowski W, Dado T, Dahbi S, Dai T, Dal Santo D, Dallapiccola C, Dam M, D'amen G, D'Amico V, Damp J, Dandoy JR, Daneri MF, Danninger M, Dao V, Darbo G, Darmora S, Das SJ, D'Auria S, David C, Davidek T, Davis-Purcell B, Dawson I, Day-Hall HA, De K, De Asmundis R, De Biase N, De Castro S, De Groot N, de Jong P, De la Torre H, De Maria A, De Salvo A, De Sanctis U, De Santo A, De Vivie De Regie JB, Dedovich DV, Degens J, Deiana AM, Del Corso F, Del Peso J, Del Rio F, Deliot F, Delitzsch CM, Della Pietra M, Della Volpe D, Dell'Acqua A, Dell'Asta L, Delmastro M, Delsart PA, Demers S, Demichev M, Denisov SP, D'Eramo L, Derendarz D, Derue F, Dervan P, Desch K, Deutsch C, Di Bello FA, Di Ciaccio A, Di Ciaccio L, Di Domenico A, Di Donato C, Di Girolamo A, Di Gregorio G, Di Luca A, Di Micco B, Di Nardo R, Diaconu C, Diamantopoulou M, Dias FA, Vale TDD, Diaz MA, Diaz Capriles FG, Didenko M, Diehl EB, Diehl L, Díez Cornell S, Diez Pardos C, Dimitriadi C, Dimitrievska A, Dingfelder J, Dinu IM, Dittmeier SJ, Dittus F, Djama F, Djobava T, Djuvsland JI, Doglioni C, Dohnalova A, Dolejsi J, Dolezal Z, Dona KM, Donadelli M, Dong B, Donini J, D'Onofrio A, D'Onofrio M, Dopke J, Doria A, Dos Santos Fernandes N, Dougan P, Dova MT, Doyle AT, Draguet MA, Dreyer E, Drivas-Koulouris I, Drnevich M, Drobac AS, Drozdova M, Du D, du Pree TA, Dubinin F, Dubovsky M, Duchovni E, Duckeck G, Ducu OA, Duda D, Dudarev A, Duden ER, D'uffizi M, Duflot L, Dührssen M, Dülsen 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    Physical review letters   Vol. 132 ( 2 ) page: 021803   2024.1

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    DOI: 10.1103/PhysRevLett.132.021803

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  76. Electrochemical Nanopipette Sensor for In Vitro/In Vivo Detection of Cu(2+) Ions.

    Timoshenko RV, Gorelkin PV, Vaneev AN, Krasnovskaya OO, Akasov RA, Garanina AS, Khochenkov DA, Iakimova TM, Klyachko NL, Abakumova TO, Shashkovskaya VS, Chaprov KD, Makarov AA, Mitkevich VA, Takahashi Y, Edwards CRW, Korchev YE, Erofeev AS

    Analytical chemistry   Vol. 96 ( 1 ) page: 127 - 136   2024.1

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    DOI: 10.1021/acs.analchem.3c03337

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  77. Early chemotherapeutic intervention to avoid thyroidectomy in pediatric Langerhans cell histiocytosis with thyroid involvement accompanying tracheal stenosis: a report of two cases.

    Aoyama S, Fukuoka K, Kubota H, Narita K, Kudo K, Mitani Y, Oshima K, Mori M, Arakawa Y, Ichimura K, Terui K, Tanami Y, Kawashima H, Nakazawa A, Niitsu T, Takahashi Y, Koh K

    International journal of hematology   Vol. 119 ( 1 ) page: 99 - 103   2024.1

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  78. Young Adult Secondary Cancer After Proton Beam Therapy: A Case Study.

    Yamamori A, Murayama S, Takahashi I, Akaihata M, Kakuda Y, Sugino T, Aramaki T, Onoe T, Takahashi Y, Ishida Y

    Advances in radiation oncology   Vol. 9 ( 1 ) page: 101307   2024.1

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  79. Modification of the association between nausea and vomiting of pregnancy and anxiety by personality trait during early pregnancy: A longitudinal study of Japanese pregnant women.

    Maruya S, Tamakoshi K, Hirose M, Takahashi Y, Yamada A, Kato N

    Japan journal of nursing science : JJNS   Vol. 21 ( 1 ) page: e12565   2024.1

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  80. Total Hydrocortisone Dosage in the Neonatal Period May Be Related to Low Developmental Quotient in Extremely Low Birth Weight Infants: A Retrospective Cohort Study.

    Taniguchi A, Nishida K, Suzuki T, Kataoka E, Fujishiro N, Kato E, Yamamoto H, Takemoto K, Ito M, Hayashi S, Sugiyama Y, Maeda T, Takahashi Y, Sato Y

    Neonatology   Vol. 121 ( 2 ) page: 195 - 202   2024

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  81. Risk factors of bloodstream infection after allogeneic hematopoietic cell transplantation in children/adolescent and young adults.

    Sajiki D, Muramatsu H, Wakamatsu M, Yamashita D, Maemura R, Tsumura Y, Imaya M, Yamamori A, Narita K, Kataoka S, Taniguchi R, Narita A, Nishio N, Takahashi Y

    PloS one   Vol. 19 ( 8 ) page: e0308395   2024

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  82. MYCN in human development and diseases.

    Nishio Y, Kato K, Oishi H, Takahashi Y, Saitoh S

    Frontiers in oncology   Vol. 14   page: 1417607   2024

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  83. Immunoglobulin-like transcript 2 as an impaired anti-tumor cytotoxicity marker of natural killer cells in patients with hepatocellular carcinoma.

    Sakata T, Yoshio S, Yamazoe T, Mori T, Kakazu E, Aoki Y, Aoyanagi N, Okamoto T, Ito T, Toyoda H, Kawaguchi T, Ono Y, Takahashi Y, Taketomi A, Kanto T

    Frontiers in immunology   Vol. 15   page: 1389411   2024

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  84. Candidate tumor-specific CD8(+) T cell subsets identified in the malignant pleural effusion of advanced lung cancer patients by single-cell analysis.

    Sugita Y, Muraoka D, Demachi-Okamura A, Komuro H, Masago K, Sasaki E, Fukushima Y, Matsui T, Shinohara S, Takahashi Y, Nishida R, Takashima C, Yamaguchi T, Horio Y, Hashimoto K, Tanaka I, Hamana H, Kishi H, Miura D, Tanaka Y, Onoue K, Onoguchi K, Yamashita Y, Stratford R, Clancy T, Yamaguchi R, Kuroda H, Ishibashi H, Okubo K, Matsushita H

    Oncoimmunology   Vol. 13 ( 1 ) page: 2371556   2024

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  85. Brain Abnormalities in Becker Muscular Dystrophy: Evaluation by Voxel-Based DTI and Morphometric Analysis.

    Maki H, Mori-Yoshimura M, Matsuda H, Hashimoto Y, Ota M, Kimura Y, Shigemoto Y, Ishihara N, Kan H, Chiba E, Arizono E, Yoshida S, Takahashi Y, Sato N

    AJNR. American journal of neuroradiology   Vol. 44 ( 12 ) page: 1405 - 1410   2023.12

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  86. Reassigning CI chondrite parent bodies based on reflectance spectroscopy of samples from carbonaceous asteroid Ryugu and meteorites.

    Amano K, Matsuoka M, Nakamura T, Kagawa E, Fujioka Y, Potin SM, Hiroi T, Tatsumi E, Milliken RE, Quirico E, Beck P, Brunetto R, Uesugi M, Takahashi Y, Kawai T, Yamashita S, Enokido Y, Wada T, Furukawa Y, Zolensky ME, Takir D, Domingue DL, Jaramillo-Correa C, Vilas F, Hendrix AR, Kikuiri M, Morita T, Yurimoto H, Noguchi T, Okazaki R, Yabuta H, Naraoka H, Sakamoto K, Tachibana S, Yada T, Nishimura M, Nakato A, Miyazaki A, Yogata K, Abe M, Okada T, Usui T, Yoshikawa M, Saiki T, Tanaka S, Terui F, Nakazawa S, Watanabe SI, Tsuda Y

    Science advances   Vol. 9 ( 49 ) page: eadi3789   2023.12

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  87. Hematological abnormalities in Jacobsen syndrome: cytopenia of varying severities and morphological abnormalities in peripheral blood and bone marrow.

    Yamashita D, Muramatsu H, Narita A, Wakamatsu M, Tsumura Y, Sajiki D, Maemura R, Yamamori A, Imaya M, Narita K, Kataoka S, Taniguchi R, Nishio N, Okuno Y, Fujita N, Koh K, Umeda K, Morihana E, Iwafuchi H, Ito M, Kojima S, Hama A, Takahashi Y

    Haematologica   Vol. 108 ( 12 ) page: 3438 - 3443   2023.12

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  88. Clinical and molecular features of CBL-mutated juvenile myelomonocytic leukemia. International journal

    Taro Yoshida, Hideki Muramatsu, Manabu Wakamatsu, Daichi Sajiki, Norihiro Murakami, Hironobu Kitazawa, Yasuhiro Okamoto, Rieko Taniguchi, Shinsuke Kataoka, Atsushi Narita, Asahito Hama, Yusuke Okuno, Yoshiyuki Takahashi

    Haematologica   Vol. 108 ( 11 ) page: 3115 - 3119   2023.11

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    DOI: 10.3324/haematol.2022.282385

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  89. T cell receptor-engineered T cells derived from target human leukocyte antigen-DPB1-specific T cell can be a potential tool for therapy against leukemia relapse following allogeneic hematopoietic cell transplantation.

    Katsuyama N, Kawase T, Barakat C, Mizuno S, Tomita A, Ozeki K, Nishio N, Sato Y, Kajiya R, Shiraishi K, Takahashi Y, Ichinohe T, Nishikawa H, Akatsuka Y

    Nagoya journal of medical science   Vol. 85 ( 4 ) page: 779 - 796   2023.11

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  90. Regulation of lipid synthesis in myelin modulates neural activity and is required for motor learning.

    Kato D, Aoyama Y, Nishida K, Takahashi Y, Sakamoto T, Takeda I, Tatematsu T, Go S, Saito Y, Kunishima S, Cheng J, Hou L, Tachibana Y, Sugio S, Kondo R, Eto F, Sato S, Moorhouse AJ, Yao I, Kadomatsu K, Setou M, Wake H

    Glia   Vol. 71 ( 11 ) page: 2591 - 2608   2023.11

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  91. Gain-of-function MYCN causes a megalencephaly-polydactyly syndrome manifesting mirror phenotypes of Feingold syndrome.

    Nishio Y, Kato K, Tran Mau-Them F, Futagawa H, Quélin C, Masuda S, Vitobello A, Otsuji S, Shawki HH, Oishi H, Thauvin-Robinet C, Takenouchi T, Kosaki K, Takahashi Y, Saitoh S

    HGG advances   Vol. 4 ( 4 ) page: 100238   2023.10

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  92. Successful treatment of DOCK8 deficiency by allogeneic hematopoietic cell transplantation from alternative donors

    Kono, A; Wakamatsu, M; Umezawa, Y; Muramatsu, H; Fujiwara, H; Tomomasa, D; Inoue, K; Hattori, K; Mitsui, T; Takada, H; Minegishi, Y; Takahashi, Y; Yamamoto, M; Mori, T; Kanegane, H

    INTERNATIONAL JOURNAL OF HEMATOLOGY   Vol. 118 ( 4 ) page: 519 - 525   2023.10

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  93. Systemic administration of clinical-grade multilineage-differentiating stress-enduring cells ameliorates hypoxic-ischemic brain injury in neonatal rats.

    Ueda K, Sato Y, Shimizu S, Suzuki T, Onoda A, Miura R, Go S, Mimatsu H, Kitase Y, Yamashita Y, Irie K, Tsuji M, Mishima K, Mizuno M, Takahashi Y, Dezawa M, Hayakawa M

    Scientific reports   Vol. 13 ( 1 ) page: 14958   2023.9

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  94. Vedolizumab for children with intestinal graft-versus-host disease: a case report and literature review

    Fukuta, T; Muramatsu, H; Yamashita, D; Sajiki, D; Maemura, R; Tsumura, Y; Yamamori, A; Imaya, M; Wakamatsu, M; Nishikawa, E; Narita, K; Kataoka, S; Taniguchi, R; Narita, A; Nishio, N; Takahashi, Y

    INTERNATIONAL JOURNAL OF HEMATOLOGY   Vol. 118 ( 3 ) page: 411 - 417   2023.9

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  95. Nanopipette Fabrication Guidelines for SICM Nanoscale Imaging.

    Takahashi Y, Sasaki Y, Yoshida T, Honda K, Zhou Y, Miyamoto T, Motoo T, Higashi H, Shevchuk A, Korchev Y, Ida H, Hanayama R, Fukuma T

    Analytical chemistry   Vol. 95 ( 34 ) page: 12664 - 12672   2023.8

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  96. Generation and purification of ACTH-secreting hPSC-derived pituitary cells for effective transplantation.

    Taga S, Suga H, Nakano T, Kuwahara A, Inoshita N, Kodani Y, Nagasaki H, Sato Y, Tsumura Y, Sakakibara M, Soen M, Miwata T, Ozaki H, Kano M, Watari K, Ikeda A, Yamanaka M, Takahashi Y, Kitamoto S, Kawaguchi Y, Miyata T, Kobayashi T, Sugiyama M, Onoue T, Yasuda Y, Hagiwara D, Iwama S, Tomigahara Y, Kimura T, Arima H

    Stem cell reports   Vol. 18 ( 8 ) page: 1657 - 1671   2023.8

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  97. Development of TCR-T cell therapy targeting mismatched HLA-DPB1 for relapsed leukemia after allogeneic transplantation.

    Barakat C, Inagaki Y, Mizuno S, Nishio N, Katsuyama N, Sato Y, Kobayashi M, Ozeki K, Iida H, Tomita A, Sawa M, Demachi-Okamura A, Takahashi Y, Nishikawa H, Akatsuka Y

    International journal of hematology   Vol. 118 ( 2 ) page: 252 - 266   2023.8

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  98. Use of the Newborn Behavioral Observations System as an early intervention for infants and their parents: A scoping review.

    Yago S, Takahashi Y, Tsukamoto E, Saito A, Saito E

    Early human development   Vol. 183   page: 105811   2023.8

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  99. Single-cell sequencing on CD8(+) TILs revealed the nature of exhausted T cells recognizing neoantigen and cancer/testis antigen in non-small cell lung cancer.

    Komuro H, Shinohara S, Fukushima Y, Demachi-Okamura A, Muraoka D, Masago K, Matsui T, Sugita Y, Takahashi Y, Nishida R, Takashima C, Ohki T, Shigematsu Y, Watanabe F, Adachi K, Fukuyama T, Hamana H, Kishi H, Miura D, Tanaka Y, Onoue K, Onoguchi K, Yamashita Y, Stratford R, Clancy T, Yamaguchi R, Kuroda H, Doi K, Iwata H, Matsushita H

    Journal for immunotherapy of cancer   Vol. 11 ( 8 )   2023.8

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  100. Skin-to-skin contact after birth: Developing a research and practice guideline.

    Brimdyr K, Stevens J, Svensson K, Blair A, Turner-Maffei C, Grady J, Bastarache L, Al Alfy A, Crenshaw JT, Giugliani ERJ, Ewald U, Haider R, Jonas W, Kagawa M, Lilliesköld S, Maastrup R, Sinclair R, Swift E, Takahashi Y, Cadwell K

    Acta paediatrica (Oslo, Norway : 1992)   Vol. 112 ( 8 ) page: 1633 - 1643   2023.8

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  101. Human leukocyte antigen 7/8-matched unrelated bone marrow transplantation using anti-thymocyte globulin in children

    Motoharu Hamada, Hideki Muramatsu, Yuka Torii, Kyogo Suzuki, Atsushi Narita, Taro Yoshida, Masayuki Imaya, Ayako Yamamori, Manabu Wakamatsu, Shunsuke Miwata, Kotaro Narita, Shinsuke Kataoka, Nozomu Kawashima, Rieko Taniguchi, Eri Nishikawa, Nobuhiro Nishio, Yoshinori Ito, Seiji Kojima, Yoshiyuki Takahashi

    International Journal of Hematology   Vol. 118 ( 1 ) page: 125 - 130   2023.7

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  102. Two Cases of Juvenile Myelomonocytic Leukemia and Myelin Oligodendrocyte Glycoprotein Antibody-Associated Disease

    Yamamoto, H; Natsume, J; Kaneko, K; Takahashi, T; Wakamatsu, M; Ogawa, C; Kumai, S; Suzui, R; Sawamura, F; Shiraki, A; Nakata, T; Kidokoro, H; Muramatsu, H; Takahashi, Y

    PEDIATRIC NEUROLOGY   Vol. 144   page: 1 - 4   2023.7

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  103. 1T/1H-SnS(2) Sheets for Electrochemical CO(2) Reduction to Formate.

    Kawabe Y, Ito Y, Hori Y, Kukunuri S, Shiokawa F, Nishiuchi T, Jeong S, Katagiri K, Xi Z, Li Z, Shigeta Y, Takahashi Y

    ACS nano   Vol. 17 ( 12 ) page: 11318 - 11326   2023.6

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  104. Successful living-donor liver transplantation for sustained liver failure even after resolution of infiltrative massive hepatic invasion of stage 4S neuroblastoma: a case report.

    Jobara K, Yamamori A, Shizuku M, Kurata N, Fujimoto Y, Muramatsu H, Takahashi Y, Ogura Y

    Surgical case reports   Vol. 9 ( 1 ) page: 100   2023.6

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  105. CLINICAL VALIDATION OF NOVEL URINARY MARKERS FOR NEUROBLASTOMA DIAGNOSIS

    Amano Hizuru, Uchida Hiroo, Harada Kazuharu, Narita Atsushi, Kumano Shun, Abe Mayumi, Sakairi Minoru, Shirota Chiyoe, Tainaka Takahisa, Sumida Wataru, Yokota Kazuki, Makita Satoshi, Osawa Tsuyoshi, Taguri Masataka, Takahashi Yoshiyuki, Hinoki Akinari

    PEDIATRIC BLOOD & CANCER   Vol. 70   page: S95 - S95   2023.6

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  106. Droplet Digital PCR Development for Adenovirus Load Monitoring in Children after Hematopoietic Stem Cell Transplantation

    Kazunori Haruta, Suguru Takeuchi, Makoto Yamaguchi, Kazuhiro Horiba, Takako Suzuki, Yuka Torii, Atsushi Narita, Hideki Muramatsu, Yoshiyuki Takahashi, Yoshinori Ito, Jun-ichi Kawada

    The Journal of Molecular Diagnostics   Vol. 25 ( 6 ) page: 403 - 409   2023.6

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  107. Dedifferentiated fat cells administration ameliorates abnormal expressions of fatty acids metabolism-related protein expressions and intestinal tissue damage in experimental necrotizing enterocolitis.

    Mimatsu H, Onoda A, Kazama T, Nishijima K, Shimoyama Y, Go S, Ueda K, Takahashi Y, Matsumoto T, Hayakawa M, Sato Y

    Scientific reports   Vol. 13 ( 1 ) page: 8266   2023.5

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  108. Germline and somatic RUNX1 variants in a pediatric bone marrow failure cohort. International journal

    Ayako Yamamori, Motoharu Hamada, Hideki Muramatsu, Manabu Wakamatsu, Asahito Hama, Atsushi Narita, Yusuke Tsumura, Taro Yoshida, Takehiko Doi, Kazuki Terada, Takeshi Higa, Nobuyuki Yamamoto, Hiroki Miura, Mitsutaka Shiota, Kenichiro Watanabe, Nao Yoshida, Ryo Maemura, Masayuki Imaya, Shunsuke Miwata, Kotaro Narita, Shinsuke Kataoka, Rieko Taniguchi, Kyogo Suzuki, Nozomu Kawashima, Nobuhiro Nishio, Hideto Iwafuchi, Masafumi Ito, Seiji Kojima, Yusuke Okuno, Yoshiyuki Takahashi

    American journal of hematology   Vol. 98 ( 5 ) page: E102 - E105   2023.5

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  109. Haematopoietic cell transplantation for children with acute megakaryoblastic leukaemia without Down syndrome

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    BRITISH JOURNAL OF HAEMATOLOGY   Vol. 201 ( 4 ) page: 747 - 756   2023.5

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  110. Graft-versus-host disease-free, relapse-free, second transplant-free survival in allogeneic hematopoietic cell transplantation for genetic disorders. International journal

    Koji Kawaguchi, Katsutsugu Umeda, Satoshi Miyamoto, Nao Yoshida, Hiromasa Yabe, Takashi Koike, Michiko Kajiwara, Hiroshi Kawaguchi, Yoshiyuki Takahashi, Masataka Ishimura, Hirotoshi Sakaguchi, Asahito Hama, Yuko Cho, Maho Sato, Keisuke Kato, Atsushi Sato, Koji Kato, Ken Tabuchi, Yoshiko Atsuta, Kohsuke Imai

    Bone marrow transplantation   Vol. 58 ( 5 ) page: 600 - 602   2023.5

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    DOI: 10.1038/s41409-023-01937-1

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  111. High-dose ubiquinol supplementation in multiple-system atrophy: a multicentre, randomised, double-blinded, placebo-controlled phase 2 trial.

    Mitsui J, Matsukawa T, Uemura Y, Kawahara T, Chikada A, Porto KJL, Naruse H, Tanaka M, Ishiura H, Toda T, Kuzuyama H, Hirano M, Wada I, Ga T, Moritoyo T, Takahashi Y, Mizusawa H, Ishikawa K, Yokota T, Kuwabara S, Sawamoto N, Takahashi R, Abe K, Ishihara T, Onodera O, Matsuse D, Yamasaki R, Kira JI, Katsuno M, Hanajima R, Ogata K, Takashima H, Matsushima M, Yabe I, Sasaki H, Tsuji S

    EClinicalMedicine   Vol. 59   page: 101920   2023.5

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    DOI: 10.1016/j.eclinm.2023.101920

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  112. Safety and efficacy of post-haematopoietic cell transplantation maintenance therapy with blinatumomab for relapsed/refractory CD19-positive B-cell acute lymphoblastic leukaemia: protocol for a phase I-II, multicentre, non-blinded, non-controlled trial (JPLSG SCT-ALL-BLIN21).

    Sakaguchi H, Umeda K, Kato I, Sakaguchi K, Hiramatsu H, Ishida H, Yabe H, Goto H, Kawahara Y, Yamashita YI, Sanada M, Deguchi T, Takahashi Y, Saito A, Noma H, Horibe K, Taga T, Adachi S, Transplantation and Cellular Therapy Committee of Japanese Childhood Cancer Group

    BMJ open   Vol. 13 ( 4 ) page: e070051   2023.4

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    DOI: 10.1136/bmjopen-2022-070051

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  113. Novel xeno-free and serum-free culturing condition to improve piggyBac transposon-based CD19 chimeric antigen receptor T-cell production and characteristics

    Sanyanusin, M; Tudsamran, S; Thaiwong, R; Tawinwung, S; Nishio, N; Takahashi, Y; Hirankarn, N; Suppipat, K

    CYTOTHERAPY   Vol. 25 ( 4 ) page: 397 - 406   2023.4

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    DOI: 10.1016/j.jcyt.2022.11.009

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  114. Efficacy and safety of bevacizumab, irinotecan, and temozolomide combination for relapsed or refractory pediatric central nervous system embryonal tumor: a single-institution study.

    Shiba Y, Motomura K, Taniguchi R, Kurimoto M, Mizutani K, Ohka F, Aoki K, Ito E, Nishikawa T, Yamaguchi J, Kibe Y, Shimizu H, Maeda S, Nakashima T, Suzuki H, Muramatsu H, Takahashi Y, Saito R

    Journal of neurosurgery. Pediatrics   Vol. 31 ( 6 ) page: 1 - 9   2023.3

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    DOI: 10.3171/2023.1.PEDS22345

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  115. Macromolecular organic matter in samples of the asteroid (162173) Ryugu.

    Yabuta H, Cody GD, Engrand C, Kebukawa Y, De Gregorio B, Bonal L, Remusat L, Stroud R, Quirico E, Nittler L, Hashiguchi M, Komatsu M, Okumura T, Mathurin J, Dartois E, Duprat J, Takahashi Y, Takeichi Y, Kilcoyne D, Yamashita S, Dazzi A, Deniset-Besseau A, Sandford S, Martins Z, Tamenori Y, Ohigashi T, Suga H, Wakabayashi D, Verdier-Paoletti M, Mostefaoui S, Montagnac G, Barosch J, Kamide K, Shigenaka M, Bejach L, Matsumoto M, Enokido Y, Noguchi T, Yurimoto H, Nakamura T, Okazaki R, Naraoka H, Sakamoto K, Connolly HC Jr, Lauretta DS, Abe M, Okada T, Yada T, Nishimura M, Yogata K, Nakato A, Yoshitake M, Iwamae A, Furuya S, Hatakeda K, Miyazaki A, Soejima H, Hitomi Y, Kumagai K, Usui T, Hayashi T, Yamamoto D, Fukai R, Sugita S, Kitazato K, Hirata N, Honda R, Morota T, Tatsumi E, Sakatani N, Namiki N, Matsumoto K, Noguchi R, Wada K, Senshu H, Ogawa K, Yokota Y, Ishihara Y, Shimaki Y, Yamada M, Honda C, Michikami T, Matsuoka M, Hirata N, Arakawa M, Okamoto C, Ishiguro M, Jaumann R, Bibring JP, Grott M, Schröder S, Otto K, Pilorget C, Schmitz N, Biele J, Ho TM, Moussi-Soffys A, Miura A, Noda H, Yamada T, Yoshihara K, Kawahara K, Ikeda H, Yamamoto Y, Shirai K, Kikuchi S, Ogawa N, Takeuchi H, Ono G, Mimasu Y, Yoshikawa K, Takei Y, Fujii A, Iijima YI, Nakazawa S, Hosoda S, Iwata T, Hayakawa M, Sawada H, Yano H, Tsukizaki R, Ozaki M, Terui F, Tanaka S, Fujimoto M, Yoshikawa M, Saiki T, Tachibana S, Watanabe SI, Tsuda Y

    Science (New York, N.Y.)   Vol. 379 ( 6634 ) page: eabn9057   2023.2

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    DOI: 10.1126/science.abn9057

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  116. Formation and evolution of carbonaceous asteroid Ryugu: Direct evidence from returned samples.

    Nakamura T, Matsumoto M, Amano K, Enokido Y, Zolensky ME, Mikouchi T, Genda H, Tanaka S, Zolotov MY, Kurosawa K, Wakita S, Hyodo R, Nagano H, Nakashima D, Takahashi Y, Fujioka Y, Kikuiri M, Kagawa E, Matsuoka M, Brearley AJ, Tsuchiyama A, Uesugi M, Matsuno J, Kimura Y, Sato M, Milliken RE, Tatsumi E, Sugita S, Hiroi T, Kitazato K, Brownlee D, Joswiak DJ, Takahashi M, Ninomiya K, Takahashi T, Osawa T, Terada K, Brenker FE, Tkalcec BJ, Vincze L, Brunetto R, Aléon-Toppani A, Chan QHS, Roskosz M, Viennet JC, Beck P, Alp EE, Michikami T, Nagaashi Y, Tsuji T, Ino Y, Martinez J, Han J, Dolocan A, Bodnar RJ, Tanaka M, Yoshida H, Sugiyama K, King AJ, Fukushi K, Suga H, Yamashita S, Kawai T, Inoue K, Nakato A, Noguchi T, Vilas F, Hendrix AR, Jaramillo-Correa C, Domingue DL, Dominguez G, Gainsforth Z, Engrand C, Duprat J, Russell SS, Bonato E, Ma C, Kawamoto T, Wada T, Watanabe S, Endo R, Enju S, Riu L, Rubino S, Tack P, Takeshita S, Takeichi Y, Takeuchi A, Takigawa A, Takir D, Tanigaki T, Taniguchi A, Tsukamoto K, Yagi T, Yamada S, Yamamoto K, Yamashita Y, Yasutake M, Uesugi K, Umegaki I, Chiu I, Ishizaki T, Okumura S, Palomba E, Pilorget C, Potin SM, Alasli A, Anada S, Araki Y, Sakatani N, Schultz C, Sekizawa O, Sitzman SD, Sugiura K, Sun M, Dartois E, De Pauw E, Dionnet Z, Djouadi Z, Falkenberg G, Fujita R, Fukuma T, Gearba IR, Hagiya K, Hu MY, Kato T, Kawamura T, Kimura M, Kubo MK, Langenhorst F, Lantz C, Lavina B, Lindner M, Zhao J, Vekemans B, Baklouti D, Bazi B, Borondics F, Nagasawa S, Nishiyama G, Nitta K, Mathurin J, Matsumoto T, Mitsukawa I, Miura H, Miyake A, Miyake Y, Yurimoto H, Okazaki R, Yabuta H, Naraoka H, Sakamoto K, Tachibana S, Connolly HC Jr, Lauretta DS, Yoshitake M, Yoshikawa M, Yoshikawa K, Yoshihara K, Yokota Y, Yogata K, Yano H, Yamamoto Y, Yamamoto D, Yamada M, Yamada T, Yada T, Wada K, Usui T, Tsukizaki R, Terui F, Takeuchi H, Takei Y, Iwamae A, Soejima H, Shirai K, Shimaki Y, Senshu H, Sawada H, Saiki T, Ozaki M, Ono G, Okada T, Ogawa N, Ogawa K, Noguchi R, Noda H, Nishimura M, Namiki N, Nakazawa S, Morota T, Miyazaki A, Miura A, Mimasu Y, Matsumoto K, Kumagai K, Kouyama T, Kikuchi S, Kawahara K, Kameda S, Iwata T, Ishihara Y, Ishiguro M, Ikeda H, Hosoda S, Honda R, Honda C, Hitomi Y, Hirata N, Hirata N, Hayashi T, Hayakawa M, Hatakeda K, Furuya S, Fukai R, Fujii A, Cho Y, Arakawa M, Abe M, Watanabe S, Tsuda Y

    Science (New York, N.Y.)   Vol. 379 ( 6634 ) page: eabn8671   2023.2

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  117. Fetal growth restriction followed by early catch-up growth impairs pancreatic islet morphology in male rats. International journal

    Mahboba Jabary, Atsuto Onoda, Yuma Kitase, Kazuto Ueda, Haruka Mimatsu, Shoji Go, Ryosuke Miura, Masahiro Tsuji, Yoshiyuki Takahashi, Masahiro Hayakawa, Yoshiaki Sato

    Scientific reports   Vol. 13 ( 1 ) page: 2732 - 2732   2023.2

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    Fetal growth restriction (FGR), followed by postnatal early catch-up growth, is associated with an increased risk of metabolic dysfunction, including type 2 diabetes in humans. This study aims to determine the effects of FGR and early catch-up growth after birth on the pathogenesis of type 2 diabetes, with particular attention to glucose tolerance, pancreatic islet morphology, and fibrosis, and to elucidate its mechanism using proteomics analysis. The FGR rat model was made by inducing mild intrauterine hypoperfusion using ameroid constrictors (ACs). On day 17 of pregnancy, ACs were affixed to the uterine and ovarian arteries bilaterally, causing a 20.9% reduction in birth weight compared to sham pups. On postnatal day 4 (P4), the pups were assigned to either the good nutrition (GN) groups with 5 pups per dam to ensure postnatal catch-up growth or poor nutrition groups with 15 pups per dam to maintain lower body weight. After weaning, all pups were fed regular chow food ad libitum (P21). Rats in both FGR groups developed glucose intolerance; however, male rats in the FGR good nutrition (FGR-GN) group also developed hypertriglyceridemia and dysmorphic pancreatic islets with fibrosis. A comprehensive and functional analysis of proteins expressed in the pancreas showed that FGR, followed by early catch-up growth, severely aggravated cell adhesion-related protein expression in male offspring. Thus, FGR and early catch-up growth caused pancreatic islet morphological abnormalities and fibrosis associated with the disturbance of cell adhesion-related protein expressions. These changes likely induce glucose intolerance and dyslipidemia in male rats.

    DOI: 10.1038/s41598-023-28584-2

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  118. 新規新生児マススクリーニングで診断し,乳児期にHLAハプロ移植を施行したムコ多糖症I型

    若松 学, 村松 秀城, 横井 克幸, 中島 葉子, 山下 大紀, 佐治木 大知, 前村 遼, 津村 悠介, 山森 彩子, 今屋 雅之, 成田 幸太郎, 谷口 理恵子, 片岡 伸介, 成田 敦, 西尾 信博, 酒井 好美, 伊藤 哲哉, 高橋 義行

    日本小児科学会雑誌   Vol. 127 ( 2 ) page: 244 - 244   2023.2

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  119. EBウイルス関連血球貪食性リンパ組織球症のシングルセルシーケンスによる病態解析

    鈴木 高子, 佐藤 好隆, 鳥居 ゆか, 福田 悠人, 春田 一憲, 山口 慎, 奥野 友介, 濱 麻人, 高橋 義行, 木村 宏, 伊藤 嘉規, 川田 潤一

    日本小児科学会雑誌   Vol. 127 ( 2 ) page: 190 - 190   2023.2

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  120. Medical records as screening tools for child death review in Japan.

    Numaguchi A, Ishii A, Natsume J, Saitoh S, Aoki Y, Yoshikawa T, Isobe I, Okumura A, Seno H, Takahashi Y

    Pediatrics international : official journal of the Japan Pediatric Society   Vol. 65 ( 1 ) page: e15692   2023.1

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    DOI: 10.1111/ped.15692

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  121. Late gastroenterological complications in children after allogeneic hematopoietic stem cell transplantation: A retrospective cohort study and a case series of children treated with Kampo medicines

    Nozomu Kawashima, Shinsuke Kataoka, Motoharu Hamada, Eri Nishikawa, Rieko Taniguchi, Atsushi Narita, Hideki Muramatsu, Nobuhiro Nishio, Yoshiyuki Takahashi

    Traditional and Kampo Medicine     2023

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    Aim: This study aimed to describe children developing late gastrointestinal (GI) complications after allogeneic hematopoietic stem cell transplantation (allo-HSCT), focusing on Kampo medicine. Methods: At the Nagoya University Hospital Department of Pediatrics, 179 consecutive patients underwent allo-HSCT over five years, of whom 167 achieved engraftment and survived at least 100 days post transplant and therefore were included in the study. Results: Thirteen patients (7.8%) developed late GI symptoms. Among these, three had upper GI symptoms, three had lower GI symptoms, while seven had upper/lower GI symptoms. Total parenteral nutrition was administered in 10 (77%) patients for a median of 81 days (range: 20–303). Four (31%) had histologically proven graft-versus-host-disease (GVHD), two (15%) had adverse drug reactions, and one (8%) had a viral infection. All three patients with lower GI symptoms responded to Kampo formulas' warming interior [TM1]. Two patients with upper GI symptoms had epigastric rigidity and responded to Aurantii Fructus Immaturus-based formulas. One patient developed severe cachexia, which was successfully reversed with Kampo medicines. There were no adverse effects due to Kampo medicines. Conclusions: Late GI complications may be a burden on children after allo-HSCT. Kampo medicines may provide a safe and promising therapy to resolve GI symptoms not otherwise specified.

    DOI: 10.1002/tkm2.1392

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  122. [Chimeric antigen receptor (CAR) T cells targeting CD19 for acute lymphoblastic leukemia].

    Takahashi Y, Nishio N

    [Rinsho ketsueki] The Japanese journal of clinical hematology   Vol. 64 ( 9 ) page: 1184 - 1191   2023

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    DOI: 10.11406/rinketsu.64.1184

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  123. Ruxolitinib in Pediatric Patients with Treatment-<i>Naive</i> or Steroid-Refractory Acute Graft-Versus-Host Disease: Primary Findings from the Phase I/II REACH4 Study

    Locatelli, F; Kang, HJ; Bruno, B; Gandemer, V; Rialland, F; Faraci, M; Takahashi, Y; Koh, K; Bittencourt, H; Cleary, G; Rosko, C; Roussou, P; St Pierre, A; Prahallad, A; Díaz-de-Heredia, C

    BLOOD   Vol. 140   page: 1376 - 1378   2022.11

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    DOI: 10.1187/blood-2022-155708

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  124. Impact of the Coronavirus Disease 2019 Pandemic on the Clinical Features of Pediatric Respiratory Syncytial Virus Infection in Japan Reviewed

    Shoko Ozeki, Jun-ichi Kawada, Daiki Yamashita, Chika Yasufuku, Takuya Akano, Masahiro Kato, Konomi Suzuki, Chihiro Tano, Kazuki Matsumoto, Shu-hei Mizutani, Ayumi Mori, Nobuhiro Nishio, Hiroyuki Kidokoro, Yoshihiro Yasui, Yoshiyuki Takahashi, Yoshiaki Sato, Anna Shiraki, Kazuto Ueda, Shotaro Ando, Noriko Nagai, Tsutomu Aoshima, Michio Suzuki, Tetsuo Kubota, Motomasa Suzuki, Satoru Doi, Daichi Fukumi, Yuichiro Sugiyama, Masafumi Morishita, Naoko Nishimura, Mizuki Takagi, Hirokazu Kurahashi, Yohei Takeuchi, Kenji Kuraishi, Osamu Shinohara, Takashi Kawabe, Nobuhiro Watanabe, Shinji Hasegawa, Taichiro Muto, Shinji Kido, Shinya Hara, Shin Hoshino

    Open Forum Infectious Diseases   Vol. 9 ( 11 ) page: ofac562   2022.11

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    Abstract

    Background

    Mitigation measures implemented during the coronavirus disease 2019 (COVID-19) pandemic remarkably reduced the incidence of infectious diseases among children. However, a re-emergence of respiratory syncytial virus (RSV) infection was observed in 2021 in Japan. We compared the clinical characteristics of hospitalized patients with RSV infection before and during COVID-19.

    Methods

    We retrospectively enrolled children aged &amp;lt;6 years who were hospitalized with RSV infection in 18 hospitals and compared their clinical characteristics before (January 2019 to April 2020, 1675 patients) and during COVID-19 (September 2020 to December 2021, 1297 patients).

    Results

    The mean age of patients with RSV infection was significantly higher during COVID-19 than before (17.4 vs 13.7 months, P &amp;lt; .001). Compared with before COVID-19, a 2.6-fold increase in RSV cases in the 2–5 years age group was observed from sentinel surveillance during COVID-19, whereas a 1.2-fold increase was noted in the same age group among hospitalized patients. On average for all patients, consolidation shadows obtained on radiography were less frequently observed (26.1 vs 29.6%, P = .04), and reduced respiratory assistance (42.2% vs 48.7%, P &amp;lt; .001) and hospitalization stay (5.7 vs 6.0 days, P &amp;lt; .001) was required in patients with RSV infection during COVID-19.

    Conclusions

    Coronavirus disease 2019 and social activity restriction caused epidemiological changes in pediatric RSV infections, and a majority of patients with RSV infection aged ≥2 years did not develop severe symptoms requiring hospitalization. The RSV symptoms during the COVID-19 outbreak were equivalent to or milder than in the previous seasons.

    DOI: 10.1093/ofid/ofac562

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    Other Link: https://academic.oup.com/ofid/article-pdf/9/11/ofac562/47043659/ofac562.pdf

  125. CHIMERIC ANTIGEN RECEPTOR T-CELL THERAPY FOLLOWED BY UR-BMT IN TYROSINE KINASE INHIBITOR RESISTANT PEDIATRIC PH1-POSITIVE ACUTE LYMPHOBLASTIC LEUKEMIA

    Imaya, M; Narita, A; Nishio, N; Wakamatsu, M; Taniguchi, R; Kataoka, S; Muramatsu, H; Ichikawa, D; Maeda, N; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  126. Impact of human leukocyte antigen mismatch on outcomes after unrelated bone marrow transplantation in paediatric patients: A retrospective analysis by the JSTCT HLA working group

    Kato, I; Sakaguchi, H; Kato, S; Sato, M; Noguchi, M; Yoshida, N; Koh, K; Koike, T; Yanagimachi, M; Kato, K; Takahashi, Y; Fujita, N; Sato, A; Hashii, Y; Tabuchi, K; Atsuta, Y; Morishima, S; Kanda, J

    BRITISH JOURNAL OF HAEMATOLOGY   Vol. 199 ( 3 ) page: 392 - 400   2022.11

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    DOI: 10.1111/bjh.18425

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  127. INFANT WITH NEUROBLASTOMA STAGE 4S REQUIRING LIVING DONOR LIVER TRANSPLANTATION

    Yamamori, A; Muramatsu, H; Wakamatsu, M; Kataoka, S; Tsuyuki, Y; Nishio, N; Shimoyama, Y; Karube, K; Ogura, Y; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  128. DANAZOL ADDED TO IMMUNOSUPPRESSIVE THERAPY IN CHILDREN WITH APLASTIC ANEMIA: A PROPENSITY SCORE-MATCHED COHORT STUDY

    Narita, A; Kudo, K; Muramatsu, H; Yagasaki, H; Kobayashi, R; Yabe, H; Ohara, A; Ohga, S; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  129. LI-FRAUMENI SYNDROME DIAGNOSED BY ONCOGENE PANEL TESTING FOR RHABDOMYOSARCOMA

    Taniguchi, R; Narita, A; Muramatsu, H; Yamashita, D; Sajiki, D; Imaya, M; Wakamatsu, M; Narita, K; Nishio, N; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  130. HEMATOLOGICAL ABNORMALITIES IN JACOBSEN SYNDROME: PATIENTS NOT ONLY DEVELOP THROMBOCYTOPENIA, BUT ALSO ANEMIA AND NEUTROPENIA

    Yamashita, D; Muramatsu, H; Narita, A; Fujita, N; Koh, K; Umeda, K; Morihana, E; Hama, A; Ito, M; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  131. TREC/KREC Newborn Screening followed by Next-Generation Sequencing for Severe Combined Immunodeficiency in Japan

    Wakamatsu, M; Kojima, D; Muramatsu, H; Okuno, Y; Kataoka, S; Nakamura, F; Sakai, Y; Tsuge, I; Ito, T; Ueda, K; Saito, A; Morihana, E; Ito, Y; Ohashi, N; Tanaka, M; Tanaka, T; Kojima, S; Nakajima, Y; Ito, T; Takahashi, Y

    JOURNAL OF CLINICAL IMMUNOLOGY   Vol. 42 ( 8 ) page: 1696 - 1707   2022.11

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    DOI: 10.1007/s10875-022-01335-0

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  132. RETROSPECTIVE NATIONWIDE SURVEY OF NONTUBERCULOUS MYCOBACTERIAL INFECTIONS IN PEDIATRIC HEMATOLOGY/ONCOLOGY PATIENTS

    Tsumura, Y; Muramatsu, H; Nishikawa, E; Tetsuka, N; Narita, A; Inoue, K; Cho, Y; Motomura, Y; Hoshino, Y; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  133. RISK FACTORS FOR BLOOD STREAM INFECTIONS AFTER ALLOGENEIC HEMATOPOIETIC CELL TRANSPLANTATION IN CHILDREN

    Sajiki, D; Muramatsu, H; Wakamatsu, M; Narita, K; Kataoka, S; Taniguchi, R; Narita, A; Nishio, N; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  134. THE DISAPPEARANCE OF MINIMAL RESIDUAL DISEASE IN BONE MARROW DEMONSTRATES GRAFT VERSUS NEUROBLASTOMA EFFECT AFTER KIR-LIGAND MISMATCHED ALLOGENEIC CBT

    Alahmadi, R; Nishio, N; Wakamatsu, M; Kataoka, S; Narita, K; Taniguchi, R; Narita, A; Muramatsu, H; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  135. TREATMENT-RELATED TOXICITY OF ANTI-GD2 ANTIBODY IMMUNOTHERAPY AFTER ALLOGENEIC STEM CELL TRANSPLANTATION IN HIGH-RISK NEUROBLASTOMA PATIENTS

    Kataoka, S; Yamamori, A; Imaya, M; Wakamatsu, M; Narita, K; Taniguchi, R; Narita, A; Muramatsu, H; Nishio, N; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  136. THREE CASES OF ABNORMAL TREC VALUES WITH COPY NUMBER ALTERATIONS IDENTIFIED IN SCID NEWBORN SCREENING PROGRAM

    Wakamatsu, M; Muramatsu, H; Kojima, D; Okuno, Y; Kataoka, S; Nakamura, T; Sakai, Y; Nakajima, Y; Ito, T; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.11

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  137. 5p deletion with congenital diaphragmatic hernia: a case report

    Kotani, T; Ushida, T; Nakamura, N; Imai, K; Iitani, Y; Tano, S; Iwagaki, S; Takahashi, Y; Ito, M; Hayakawa, M; Kajiyama, H

    JOURNAL OF MEDICAL CASE REPORTS   Vol. 16 ( 1 ) page: 390   2022.10

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    DOI: 10.1186/s13256-022-03579-1

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  138. Involvement of the Thalamus, Hippocampus, and Brainstem in Hypsarrhythmia of West Syndrome: Simultaneous Recordings of Electroencephalography and fMRI Study

    Maki, Y; Natsume, J; Ito, Y; Okai, Y; Bagarinao, E; Yamamoto, H; Ogaya, S; Takeuchi, T; Fukasawa, T; Sawamura, F; Mitsumatsu, T; Maesawa, S; Saito, R; Takahashi, Y; Kidokoro, H

    AMERICAN JOURNAL OF NEURORADIOLOGY   Vol. 43 ( 10 ) page: 1502 - 1507   2022.10

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  139. 深層学習を用いた新生児頭部エコー画像による脳室周囲白質軟化症の発症予測

    川口 将宏, 城所 博之, 白木 杏奈, 山本 啓之, 中田 智彦, 夏目 淳, 高橋 義行, 伊藤 倫太郎, 野田 晴香, 前田 剛志, 上田 一仁, 伊藤 美春, 佐藤 義朗, 早川 昌弘

    日本小児科学会雑誌   Vol. 126 ( 10 ) page: 1445 - 1445   2022.10

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  140. Quantitative assessment of viral load in the blood and urine of patients with congenital cytomegalovirus infection using droplet digital PCR

    Yamaguchi, M; Kawada, J; Torii, Y; Haruta, K; Suzuki, T; Horiba, K; Takahashi, Y; Ito, Y

    JOURNAL OF MEDICAL VIROLOGY   Vol. 94 ( 9 ) page: 4559 - 4564   2022.9

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    DOI: 10.1002/jmv.27844

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  141. Clinical parameter-based prediction of DNA methylation classification generates a prediction model of prognosis in patients with juvenile myelomonocytic leukemia

    Imaizumi, T; Meyer, J; Wakamatsu, M; Kitazawa, H; Murakami, N; Okuno, Y; Yoshida, T; Sajiki, D; Hama, A; Kojima, S; Takahashi, Y; Loh, M; Stieglitz, E; Muramatsu, H

    SCIENTIFIC REPORTS   Vol. 12 ( 1 ) page: 14753   2022.8

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    DOI: 10.1038/s41598-022-18733-4

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  142. Whole-exome analysis of 177 pediatric patients with undiagnosed diseases. International journal

    Kotaro Narita, Hideki Muramatsu, Satoshi Narumi, Yuji Nakamura, Yusuke Okuno, Kyogo Suzuki, Motoharu Hamada, Naoya Yamaguchi, Atsushi Suzuki, Yosuke Nishio, Anna Shiraki, Ayako Yamamori, Yusuke Tsumura, Fumi Sawamura, Masahiro Kawaguchi, Manabu Wakamatsu, Shinsuke Kataoka, Kohji Kato, Hideyuki Asada, Tetsuo Kubota, Yukako Muramatsu, Hiroyuki Kidokoro, Jun Natsume, Seiji Mizuno, Tomohiko Nakata, Hidehito Inagaki, Naoko Ishihara, Takahiro Yonekawa, Akihisa Okumura, Tomoo Ogi, Seiji Kojima, Tadashi Kaname, Tomonobu Hasegawa, Shinji Saitoh, Yoshiyuki Takahashi

    Scientific reports   Vol. 12 ( 1 ) page: 14589 - 14589   2022.8

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    Recently, whole-exome sequencing (WES) has been used for genetic diagnoses of patients who remain otherwise undiagnosed. WES was performed in 177 Japanese patients with undiagnosed conditions who were referred to the Tokai regional branch of the Initiative on Rare and Undiagnosed Diseases (IRUD) (TOKAI-IRUD). This study included only patients who had not previously received genome-wide testing. Review meetings with specialists in various medical fields were held to evaluate the genetic diagnosis in each case, which was based on the guidelines of the American College of Medical Genetics and Genomics. WES identified diagnostic single-nucleotide variants in 66 patients and copy number variants (CNVs) in 11 patients. Additionally, a patient was diagnosed with Angelman syndrome with a complex clinical phenotype upon detection of a paternally derived uniparental disomy (UPD) [upd(15)pat] wherein the patient carried a homozygous DUOX2 p.E520D variant in the UPD region. Functional analysis confirmed that this DUOX2 variant was a loss-of-function missense substitution and the primary cause of congenital hypothyroidism. A significantly higher proportion of genetic diagnoses was achieved compared to previous reports (44%, 78/177 vs. 24-35%, respectively), probably due to detailed discussions and the higher rate of CNV detection.

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  143. Microsatellite instability-high is rare events in refractory pediatric solid tumors. International journal

    Taro Yoshida, Hideki Muramatsu, Manabu Wakamatsu, Rieko Taniguchi, Daisuke Ichikawa, Masato Nakaguro, Atsushi Natsume, Yoshiyuki Takahashi

    Pediatric hematology and oncology   Vol. 39 ( 5 ) page: 468 - 474   2022.8

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    Microsatellite instability (MSI)-high status is associated with good responsiveness to immune checkpoint inhibitors. Although MSI-high status has been actively investigated in pediatric brain tumors, studies of other pediatric solid tumors are lacking. Among 334 consecutive pediatric patients with solid tumors, we retrospectively analyzed formalin-fixed paraffin-embedded tumor tissues of 36 of 74 patients (49%) who died of disease. We assessed the MSI status in these tissues using five multiplexed markers. The results revealed that none of the patients had an MSI-high status. These results indicate that MSI-high status is a rare event in pediatric patients with refractory/relapsed solid tumors.Supplemental data for this article is available online at https://doi.org/10.1080/08880018.2021.1998266.

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  144. Prognostic factors of children and adolescents with T-cell acute lymphoblastic leukemia after allogeneic transplantation. International journal

    Hisashi Ishida, Motohiro Kato, Yuta Kawahara, Sae Ishimaru, Yuho Najima, Shinichi Kako, Maho Sato, Mitsuteru Hiwatari, Maiko Noguchi, Keisuke Kato, Katsuyoshi Koh, Keiko Okada, Fuminori Iwasaki, Ryoji Kobayashi, Shunji Igarashi, Shoji Saito, Yoshiyuki Takahashi, Atsushi Sato, Junji Tanaka, Yoshiko Hashii, Yoshiko Atsuta, Hirotoshi Sakaguchi, Toshihiko Imamura

    Hematological oncology   Vol. 40 ( 3 ) page: 457 - 468   2022.8

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    Acute lymphoblastic leukemia (ALL) is the most common cancer during childhood, and some high-risk patients with ALL require hematopoietic stem cell transplantation (HSCT). Mainly due to small patient numbers, studies focusing specifically on children and adolescents with T-cell ALL (T-ALL) are limited. Using a nationwide registry, we retrospectively analyzed data from patients under 20 years old who underwent their first HSCT for T-ALL between 2000 and 2018. As a result, total 484 patients were included, and their median follow-up period was 6.9 years after HSCT for survivors. While patients receiving HSCT at first complete remission (CR) showed relatively good 5-year leukemia free survival (5yLFS, 73.5%), once relapse occurred, their prognosis was much worse (44.4%) even if they attained second remission again (p < 0.001). Among patients receiving HSCT at CR1, grade II-IV acute graft versus host disease was associated with worse overall and LFS than grade 0-I (5yLFS 69.5% vs. 82.1%, p = 0.026) mainly due to high non-relapse mortality. Among those patients, patients receiving related bone marrow transplantation, unrelated bone marrow transplantation, or unrelated cord blood transplantation showed similar survival (5yLFS, 73.2%, 76.3%, and 77.0%, respectively). For patients undergoing cord blood transplantation at CR1, total-body irradiation-based myeloablative conditioning was associated with better 5yLFS than other conditioning regimens (85.4% vs. 62.2%, p = 0.044), as it reduced the risk of relapse. These results indicate that relapsed patients have much less chance of cure, and that identifying patients who require HSCT for cure and offering them HSCT with optimal settings during CR1 are crucial for children and adolescents with T-ALL.

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  145. Risk factors for unplanned removal of central venous catheters in hospitalized children with hematological and oncological disorders.

    Moe Miyagishima, Motoharu Hamada, Yuji Hirayama, Hideki Muramatsu, Takahisa Tainaka, Chiyoe Shirota, Akinari Hinoki, Takahiro Imaizumi, Masahiro Nakatochi, Michi Kamei, Eri Nishikawa, Nozomu Kawashima, Atsushi Narita, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    International journal of hematology   Vol. 116 ( 2 ) page: 288 - 294   2022.8

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    Central venous catheters (CVCs) are essential devices in the treatment of pediatric patients with hematological and oncological disorders; however, the most suitable type of CVC for these patients remains unclear. We retrospectively compared risk factors for unplanned removal of two commonly used CVCs, peripherally inserted central catheters (PICCs) and tunneled CVCs, to propose which is the better device. We followed 89 patients fitted with a tunneled CVC (total 21,395 catheter-days) and 84 fitted with a PICC (total 9177 catheter-days) between January 1, 2013 and December 31, 2015, until catheter removal. Patients with a PICC had a significantly higher 3-month cumulative incidence of catheter occlusion (5.2% vs. 0%, p = 4.08 × 10-3) and total unplanned removals (29.0% vs. 6.9%, p = 0.0316) than those with tunneled CVCs. However, the cumulative incidence of central line-associated bloodstream infection did not differ significantly by CVC type. Multivariable analysis identified younger age (< 2 years) [sub-distribution hazard ratio (SHR) 2.29; 95% confidence interval (CI) 1.27-4.14] and PICC (SHR 2.73; 95% CI 1.48-5.02) as independent risk factors for unplanned removal. Thus, our results suggest that tunneled CVCs are preferable in pediatric patients with hematological and oncological disorders requiring long-term, intensive treatment.

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  146. 愛知県における重症複合免疫不全症に対するTREC新生児マススクリーニング検査

    若松 学, 村松 秀城, 小島 大英, 奥野 友介, 片岡 伸介, 成田 敦, 中島 葉子, 柘植 郁哉, 酒井 好美, 伊藤 哲哉, 高橋 義行

    臨床血液   Vol. 63 ( 8 ) page: 957 - 957   2022.8

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  147. A patient with very early onset FH-deficient renal cell carcinoma diagnosed at age seven. International journal

    Rieko Taniguchi, Hideki Muramatsu, Yusuke Okuno, Taro Yoshida, Manabu Wakamatsu, Motoharu Hamada, Chiyoe Shirota, Wataru Sumida, Akinari Hinoki, Takahisa Tainaka, Yoshimitsu Gotoh, Toyonori Tsuzuki, Yukichi Tanaka, Seiji Kojima, Hiroo Uchida, Yoshiyuki Takahashi

    Familial cancer   Vol. 21 ( 3 ) page: 337 - 341   2022.7

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    Hereditary leiomyomatosis and renal cell cancer (HLRCC) is caused by heterozygous germline variants in the fumarate hydratase (FH) gene and is associated with increased susceptibility to cutaneous leiomyomas, uterine leiomyomas, and renal cell carcinoma (RCC). HLRCC-associated RCC usually occurs in the middle age, with the median age being 40-44 years. This report describes a seven-year-old (84-month-old) male who developed a large right kidney tumor with multiple cystic lesions that contained enhanced solid components. There was no evidence of distant metastasis. The male patient underwent right nephrectomy and has been recovering well without metastasis or recurrence. Pathological examination revealed that tumor cells with relatively prominent nucleoli and surrounded by halos, were located in a limited area. Immunohistochemical staining was negative for FH. Whole-exome sequencing identified his germline variant in the FH gene and its loss of heterozygosity in the tumor. At nine years (114 months) of age, the male patient showed no recurrence of the tumor. This was the youngest-onset case of HLRCC-associated RCC to date. This report may affect the starting age for future RCC-surveillance programs for patients with HLRCC.

    DOI: 10.1007/s10689-021-00268-8

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  148. Difficulties in diagnosing Malassezia furfur bloodstream infection and possibility of spontaneous resolution in a patient undergoing chemotherapy for neuroblastoma: A case report. International journal

    Nobuyuki Tetsuka, Hideki Muramatsu, Mitsutaka Iguchi, Keisuke Oka, Hiroshi Morioka, Yoshiyuki Takahashi, Tetsuya Yagi

    Journal of infection and chemotherapy : official journal of the Japan Society of Chemotherapy   Vol. 28 ( 7 ) page: 987 - 990   2022.7

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    Malassezia furfur is a lipophilic, yeast-like fungus that forms part of the normal human skin microflora and is associated with a wide range of infections, such as pityriasis versicolor, folliculitis, and systemic infections in immunocompromised patients. Although matrix-assisted laser desorption/ionization time-of-flight mass spectrometry has enabled rapid identification of Malassezia species, it is still a challenge to diagnose systemic infections because Malassezia fungemia can often be missed by automated blood culture systems. We report a case in which M. furfur in blood was detected by the presence of yeast-like fungi in blood smears. Yeast-like organisms were observed in the blood smears of a 3-year-old boy, taken over 2 weeks without any symptoms. He had undergone several courses of chemotherapy for neuroblastoma via an indwelling central venous catheter (CVC) that was placed in his right anterior chest for 11 months. Although the blood cultures obtained from an automated blood culture system were negative, M. furfur growth was detected in the subcultured blood taken from the CVC. The CVC was removed, and the scheduled chemotherapy was postponed. No systemic M. furfur bloodstream infection occurred; the infection resolved spontaneously without any specific treatment; only prophylactic fluconazole was administered. M. furfur fungemia may not be diagnosable by an automated blood culture system. Further, M. furfur may not cause infections in humans even when administered intravenously. This report may lead to the discovery of factors related to human infectivity of this disease in the future.

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  149. Prognostic Factors for Outcomes of Allogeneic HSCT for Children and Adolescents/Young Adults With CML in the TKI Era. International journal

    Hiroyuki Shimada, Akihiko Tanizawa, Takeshi Kondo, Tokiko Nagamura-Inoue, Masahiro Yasui, Arinobu Tojo, Hideki Muramatsu, Tetsuya Eto, Noriko Doki, Masatsugu Tanaka, Maho Sato, Maiko Noguchi, Naoyuki Uchida, Yoshiyuki Takahashi, Naoki Sakata, Tatsuo Ichinohe, Yoshiko Hashii, Koji Kato, Yoshiko Atsuta, Kazuteru Ohashi

    Transplantation and cellular therapy   Vol. 28 ( 7 ) page: 376 - 389   2022.7

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    The breakthrough effects of tyrosine kinase inhibitors (TKIs) have lessened indications for allogeneic hematopoietic stem cell transplantation (HSCT) in chronic myeloid leukemia (CML). However, HSCT is still attractive for children and adolescents/young adults (AYAs) requiring lifelong TKI therapy. Nevertheless, little has been reported on the outcomes of large clinical studies of HSCT targeting these age groups. This study aimed to identify prognostic factors for the outcomes of HSCT, including reduced-intensity conditioning (RIC)-HSCT, for children and AYAs with CML in the TKI era. We performed a registry analysis for 200 patients with CML aged <30 years who underwent pretransplant TKI therapy from the observational nationwide database established by the Japanese Society for Transplantation and Cellular Therapy. The patients received bone marrow (BM), peripheral blood (PB), or cord blood (CB) from either related or unrelated donors. The indication for HSCT for individual patients was determined by the institution according to European LeukemiaNet recommendations and other guidelines. The 5-year overall survival (OS) rates for patients with chronic phase (CP) (n = 124), accelerated phase (AP) (n = 23), and blastic phase (BP) (n = 53) at diagnosis were 82.8%, 71.1%, and 73.3%, respectively, with no significant difference (P =.3293). The strongest predictor of engraftment was transplant source, with CB (hazard ratio [HR], 0.33) and PB (HR, 2.00) (compared with BM) being independent unfavorable and favorable predictors, respectively. Transplant source was also an independent predictor of chronic GVHD, with PB (HR, 1.81) and CB (HR, 0.39) (compared with BM) being unfavorable and favorable predictors, respectively. The strongest predictor of OS rate for patients with CP at diagnosis was disease phase at HSCT, with second or greater CP, AP, or BP (HR, 2.81) (compared with first CP [CP1]) being an unfavorable predictor. In addition, patients with CP at diagnosis who had major and complete molecular responses at HSCT had excellent outcomes, with 5-year OS rates of 100% and 94.4%, respectively. The 5-year OS rate was compared between RIC (n = 31) and myeloablative conditioning (MAC) (n = 58) in patients with CP1, both of which were 89.3%, with no significant difference (P = .9440). On univariate analysis for the RIC cohort with CP at diagnosis, the age at HSCT (HR, 1.27) (increase per year) and the time from diagnosis to HSCT (HR, 1.83) (increase per year) were significant predictors for OS. Our study demonstrates that RIC may be an appropriate alternative to MAC for children and AYAs with CP1. As for the transplant source, we recommend first selecting BM because of a higher engraftment rate compared to CB and a lower incidence of chronic GVHD compared to PB. Although HSCT in the status of a major molecular response is desirable, it is not advisable to continue TKI pointlessly long because age at HSCT and timing of HSCT are prognostic factors that determine survival. The decision to perform RIC-HSCT instead of continuing TKI should be carefully made, considering the possibility of transplant-related complications.

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  150. [Recommendations (Proposal) for promoting research for overcoming neurological diseases 2020].

    Mochizuki H, Aoki M, Ikenaka K, Inoue H, Iwatsubo T, Ugawa Y, Okazawa H, Ono K, Onodera O, Kitagawa K, Saito Y, Shimohata T, Takahashi R, Toda T, Nakahara J, Matsumoto R, Mizusawa H, Mitsui J, Murayama S, Katsuno M, Future Vision Committee of Japanese Society of Neurology, Aoki Y, Ishiura H, Izumi Y, Koike H, Shimada H, Takahashi Y, Tokuda T, Nakajima H, Hatano T, Misawa S, Watanabe H

    Rinsho shinkeigaku = Clinical neurology   Vol. 62 ( 6 ) page: 443 - 457   2022.6

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    DOI: 10.5692/clinicalneurol.cn-001696

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  151. [Recommendations (Proposal) for promoting research for overcoming neurological diseases 2020].

    Mochizuki H, Aoki M, Ikenaka K, Inoue H, Iwatsubo T, Ugawa Y, Okazawa H, Ono K, Onodera O, Kitagawa K, Saito Y, Shimohata T, Takahashi R, Toda T, Nakahara J, Matsumoto R, Mizusawa H, Mitsui J, Murayama S, Katsuno M, Future Vision Committee of Japanese Society of Neurology, Aoki Y, Ishiura H, Izumi Y, Koike H, Shimada H, Takahashi Y, Tokuda T, Nakajima H, Hatano T, Misawa S, Watanabe H

    Rinsho shinkeigaku = Clinical neurology   Vol. 62 ( 6 ) page: 429 - 442   2022.6

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    DOI: 10.5692/clinicalneurol.cn-001695

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  152. Registry data analysis of hematopoietic stem cell transplantation on systemic chronic active Epstein-Barr virus infection patients in Japan

    Yamamoto, M; Sato, M; Onishi, Y; Sasahara, Y; Sano, H; Masuko, M; Nakamae, H; Matsuoka, KI; Ara, T; Washio, K; Onizuka, M; Watanabe, K; Takahashi, Y; Hirakawa, T; Nishio, M; Sakashita, C; Kobayashi, T; Sawada, A; Ichinohe, T; Fukuda, T; Hashii, Y; Atsuta, Y; Arai, A

    AMERICAN JOURNAL OF HEMATOLOGY   Vol. 97 ( 6 ) page: 780 - 790   2022.6

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    DOI: 10.1002/ajh.26544

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  153. METABOLOMICS OF URINARY METABOLITES IN CHILDHOOD RHABDOMYOSARCOMA TO DISCOVER NOVEL BIOMARKERS

    Nakano, S; Uchida, H; Amano, H; Narita, A; Abe, M; Ishigaki, T; Sakairi, M; Shirota, C; Sumida, W; Makita, S; Takimoto, A; Okamoto, M; Yasui, A; Takada, S; Nakagawa, Y; Terui, Y; Sunagawa, M; Takahashi, Y; Osawa, T; Hinoki, A

    PEDIATRIC BLOOD & CANCER   Vol. 69   2022.6

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  154. TREC測定による新生児マススクリーニングを通じて診断されたX連鎖重症複合免疫不全症の1例

    山下 大紀, 村松 秀城, 佐治木 大知, 津村 悠介, 前村 遼, 今屋 雅之, 山森 彩子, 若松 学, 片岡 伸介, 濱田 太立, 谷口 理恵子, 川島 希, 西川 英里, 成田 敦, 奥野 友介, 西尾 信博, 小島 大英, 中島 葉子, 柘植 郁哉, 中村 富美子, 酒井 好美, 伊藤 哲哉, 高橋 義行

    日本小児科学会雑誌   Vol. 126 ( 6 ) page: 970 - 970   2022.6

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  155. Combination chemotherapy consisting of irinotecan, etoposide, and carboplatin for refractory or relapsed neuroblastoma. International journal

    Masayuki Imaya, Hideki Muramatsu, Atsushi Narita, Ayako Yamamori, Manabu Wakamatsu, Taro Yoshida, Shunsuke Miwata, Kotaro Narita, Daisuke Ichikawa, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    Cancer medicine   Vol. 11 ( 9 ) page: 1956 - 1964   2022.5

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    BACKGROUND: Patients with primary refractory and relapsed neuroblastoma have a poor prognosis since safe and effective chemotherapies for these patients are currently limited. The development of new chemotherapy regimens for these patients is imperative to improve survival outcomes. METHODS: We retrospectively analyzed 40 patients with refractory (n = 36) or relapsed (n = 4) neuroblastoma who received irinotecan, etoposide, and carboplatin (IREC) as a second-line treatment. We evaluated their therapeutic response and the toxicity of IREC. We also assessed the impact of UGT1A1 gene polymorphisms, which are involved in irinotecan metabolism, on outcomes and toxicity. RESULTS: A total of 112 cycles of IREC were administered to 40 patients with a median of 2 cycles per patient (range, 1-9). Six (15%) patients (UGT1A1 wild-type [n = 2] and heterozygous [n = 4]) showed objective responses, including partial response (n = 1), tumor shrinkage (n = 4), and improved findings on their MIBG scan (n = 1). Grade 4 neutropenia, grade 4 leukopenia, and grades 3-4 gastrointestinal toxicity were observed in 110 (98%), 88 (79%), and 3 (3%) cycles, respectively. There was no IREC-related mortality. Patients with UGT1A1 polymorphisms showed a higher frequency of grade 4 leukopenia, but these patients did not have increased treatment-related mortality or non-hematologic toxicity. CONCLUSIONS: IREC showed an objective response rate of 15% including 1 case with partial response. IREC was well tolerated regardless of UGT1A1 genotype. This study suggests that IREC is a promising second-line chemotherapy for refractory or relapsed neuroblastoma.

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  156. Predictive factors of acute respiratory events during initial induction chemotherapy in patients with advanced neuroblastoma

    Matsui, M; Makimoto, A; Nishio, N; Takahashi, Y; Urashima, M; Yuza, Y

    CANCER REPORTS   Vol. 5 ( 5 ) page: e1499   2022.5

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    DOI: 10.1002/cnr2.1499

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  157. 脊髄圧迫によるoncologic emergencyを呈した大腰筋内の後腹膜腫瘍

    片岡 伸介, 佐治木 大知, 津村 悠介, 前村 遼, 今屋 雅之, 山森 彩子, 若松 学, 谷口 理恵子, 濱田 太立, 西川 英里, 川島 希, 成田 敦, 村松 秀城, 西尾 信博, 高橋 義行, 内田 広夫, 中川 洋一, 田井中 貴久, 安井 昭洋, 中黒 匡人, 下山 芳江

    日本小児血液・がん学会雑誌   Vol. 59 ( 1 ) page: 72 - 72   2022.5

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  158. Minor PNH clones do not distinguish inherited bone marrow failure syndromes from immune-mediated aplastic anemia. International journal

    Atsushi Narita, Shunsuke Miwata, Masayuki Imaya, Yusuke Tsumura, Ayako Yamamori, Manabu Wakamatsu, Motoharu Hamada, Rieko Taniguchi, Yusuke Okuno, Hideki Muramatsu, Yoshiyuki Takahashi

    Blood advances   Vol. 6 ( 8 ) page: 2517 - 2519   2022.4

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    DOI: 10.1182/bloodadvances.2021006044

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  159. Death review of children receiving medical care at home. International journal

    Jun Natsume, Atsushi Numaguchi, Atsuko Ohno, Mihoko Mizuno, Yoshiyuki Takahashi, Akihisa Okumura, Tetsushi Yoshikawa, Shinji Saitoh, Kiyokuni Miura, Masaharu Noda

    Pediatric research   Vol. 91 ( 5 ) page: 1286 - 1289   2022.4

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    BACKGROUND: Children receiving home medical care need special attention to prevent unexpected death. The aim of this study was to clarify the factors contributing to death in children receiving home medical care from the child death review database. METHODS: Children receiving home medical care were enrolled from the child death review database from 2014 to 2016 in Aichi prefecture, Japan, with a population of one million children. Types of medical care and factors contributing to death were examined. RESULTS: Of the 631 children who died, 40 children (6%) were receiving home medical care (21: tracheostomy; 19: ventilator; 26: suctioning of naso-oral secretions; 19: oxygen inhalation; 32: tube feeding; 6: urethral catheterization; and 1: peritoneal dialysis). The death rate was 50 times that in the general population of children. Ten children had contributory factors that seemed to be preventable. In four children, the families could not replace the tracheostomy tubes during an accident. In three, oxygen saturation or ventilator alarms were not set appropriately. In two, an oxygen cylinder became empty. One child fell down from a seat in a car. CONCLUSIONS: Improvement of devices and correct guidance to caregivers may reduce the death rate in children receiving home medical care. IMPACT: Children receiving home medical care, such as tracheostomy care, mechanical ventilation, or tube feeding, need special attention to prevent unexpected death. In this population-based child death review, 6% of children received home medical care, and it was estimated that 1 of 100 children receiving home medical care died per year. One-quarter of the deaths could be preventable by caregiver education or development of devices.

    DOI: 10.1038/s41390-021-01606-3

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  160. Hematopoietic Cell Transplantation for Inborn Errors of Immunity Other than Severe Combined Immunodeficiency in Japan: Retrospective Analysis for 1985-2016. International journal

    Satoshi Miyamoto, Katsutsugu Umeda, Mio Kurata, Masakatsu Yanagimachi, Akihiro Iguchi, Yoji Sasahara, Keiko Okada, Takashi Koike, Reo Tanoshima, Masataka Ishimura, Masafumi Yamada, Maho Sato, Yoshiyuki Takahashi, Michiko Kajiwara, Hiroshi Kawaguchi, Masami Inoue, Yoshiko Hashii, Hiromasa Yabe, Koji Kato, Yoshiko Atsuta, Kohsuke Imai, Tomohiro Morio

    Journal of clinical immunology   Vol. 42 ( 3 ) page: 529 - 545   2022.4

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    PURPOSE: Hematopoietic cell transplantation (HCT) is a curative therapy for most patients with inborn errors of immunity (IEI). We conducted a nationwide study on HCT for patients with IEI other than severe combined immunodeficiency (non-SCID) in Japan. METHODS: Data from the Japanese national database (Transplant Registry Unified Management Program, TRUMP) for 566 patients with non-SCID IEI, who underwent their first HCT between 1985 and 2016, were retrospectively analyzed. RESULTS: The 10-year overall survival (OS) and event-free survival (EFS) were 74% and 64%, respectively. The 10-year OS for HCT from unrelated bone marrow (URBM), accounting for 39% of HCTs, was comparable to that for HCT from matched sibling donor (MSD), 79% and 81%, respectively. HCT from unrelated cord blood (URCB), accounting for 28% of HCTs, was also common, with a 10-year OS of 69% but less robust engraftment. The intensity of conditioning was not associated with OS or neutrophil recovery; however, myeloablative conditioning was more frequently associated with infection-related death. Patients who received myeloablative irradiation showed poor OS. Multivariate analyses revealed that HCT in 1985-1995 (hazard ratio [HR], 2.0; P = 0.03), URCB (HR, 2.0; P = 0.01), and related donor other than MSD (ORD) (HR, 2.9; P < 0.001) were associated with poor OS, and URCB (HR, 3.6; P < 0.001) and ORD (HR, 2.7; P = 0.02) showed a higher incidence of retransplantation. CONCLUSIONS: We present the 1985-2016 status of HCT for non-SCID IEI in Japan with sufficient statistical power, highlighting the potential of URBM as an alternative donor and the feasibility of reduced intensity conditioning.

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  161. Genetic Study of Fanconi Anemia in Infancy Revealed <i>FANCI</i> Mutations and Defective <i>ALDH2</i> Variant: A Case Report

    Urata, T; Imamura, T; Osone, S; Muramatsu, H; Takahashi, Y; Hosoi, H

    JOURNAL OF PEDIATRIC HEMATOLOGY ONCOLOGY   Vol. 44 ( 2 ) page: E438 - E441   2022.3

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    DOI: 10.1097/MPH.0000000000002254

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  162. Short Report Nosocomial infection with rotavirus vaccine strain in paediatric patients with immunodeficiency

    Miura, H; Taniguchi, K; Narita, K; Kawamura, Y; Kozawa, K; Muramatsu, H; Takahashi, Y; Ihira, M; Yoshikawa, T

    JOURNAL OF HOSPITAL INFECTION   Vol. 121   page: 9 - 13   2022.3

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    DOI: 10.1016/j.jhin.2021.12.009

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  163. Prospective validation of the provisional entity of refractory cytopenia of childhood, proposed by the World Health Organization

    Hama, A; Hasegawa, D; Manabe, A; Nozawa, K; Narita, A; Muramatsu, H; Kosaka, Y; Kobayashi, M; Koh, K; Takahashi, Y; Watanabe, K; Ohara, A; Ito, M; Kojima, S

    BRITISH JOURNAL OF HAEMATOLOGY   Vol. 196 ( 4 ) page: 1031 - 1039   2022.2

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    DOI: 10.1111/bjh.17921

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  164. Targeting MEF2D-fusion Oncogenic Transcriptional Circuitries in B-cell Precursor Acute Lymphoblastic Leukemia

    Tsuzuki, S; Yasuda, T; Kawazu, M; Ueno, T; Karnan, S; Ota, A; Sanada, M; Nagai, H; Tomita, A; Takahashi, Y; Miyazaki, Y; Matsumura, I; Kiyoi, H; Hosokawa, Y; Mano, H; Hayakawa, F

    CANCER SCIENCE   Vol. 113   page: 1236 - 1236   2022.2

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  165. Dyserythropoietic anaemia with an intronic GATA1 splicing mutation in patients suspected to have Diamond-Blackfan anaemia. International journal

    Akie Kobayashi, Ryusei Ohtaka, Tsutomu Toki, Junichi Hara, Hideki Muramatsu, Rika Kanezaki, Yuka Takahashi, Tomohiko Sato, Takuya Kamio, Ko Kudo, Shinya Sasaki, Taro Yoshida, Taiju Utsugisawa, Hitoshi Kanno, Kenichi Yoshida, Yasuhito Nannya, Yoshiyuki Takahashi, Seiji Kojima, Satoru Miyano, Seishi Ogawa, Kiminori Terui, Etsuro Ito

    EJHaem   Vol. 3 ( 1 ) page: 163 - 167   2022.2

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    Diamond-Blackfan anaemia (DBA) shares clinical features with two recently reported sporadic cases of dyserythropoietic anaemia with a cryptic GATA1 splicing mutation (c.871-24 C>T). We hypothesized that some patients clinically diagnosed with DBA but whose causative genes were unknown may carry the intronic GATA1 mutation. Here, we examined 79 patients in our DBA cohort, who had no detectable causative genes. The intronic GATA1 mutation was identified in two male patients sharing the same pedigree that included multiple cases with anaemia. Cosegregation of this mutation and disease in multiple family members provide evidence to support the pathogenicity of the intronic GATA1 mutation.

    DOI: 10.1002/jha2.374

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  166. 胎児発育不全モデルラットの脳脊髄液中タンパク質の網羅的解析 脳発達異常の病態解明に向けて

    小野田 淳人, 北瀬 悠磨, 辻 雅弘, 上田 一仁, 高橋 義行, 早川 昌弘, 佐藤 義朗

    日本小児科学会雑誌   Vol. 126 ( 2 ) page: 414 - 414   2022.2

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  167. 投与経路に着目したMultilineage-differentiating stress enduring cell(Muse細胞)を用いた新生児慢性肺疾患の新規治療法開発

    三浦 良介, 佐藤 義朗, 小野田 淳人, 藤本 健志, 後藤 良子, 高本 幸奈, 神澤 孝洋, 鈴木 紗記子, 上田 一仁, 徐 悦, 橋本 佑樹, 落合 加奈代, 谷口 顕信, 田中 龍一, 伊藤 美春, 齊藤 明子, 村松 友佳子, 清水 忍, 高橋 義行, 早川 昌弘

    日本小児科学会雑誌   Vol. 126 ( 2 ) page: 250 - 250   2022.2

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  168. TREC測定による新生児マススクリーニングにより診断され同種臍帯血移植を行ったX連鎖重症複合免疫不全症

    山下 大紀, 村松 秀城, 今屋 雅之, 山森 彩子, 若松 学, 片岡 伸介, 濱田 太立, 谷口 理恵子, 川島 希, 西川 英里, 成田 敦, 奥野 友介, 西尾 信博, 小島 大英, 中島 葉子, 柘植 郁哉, 中村 冨美子, 酒井 好美, 伊藤 哲哉, 高橋 義行

    日本小児科学会雑誌   Vol. 126 ( 2 ) page: 281 - 281   2022.2

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  169. TREC測定による新生児マススクリーニングにより診断され同種臍帯血移植を行ったX連鎖重症複合免疫不全症

    山下 大紀, 村松 秀城, 今屋 雅之, 山森 彩子, 若松 学, 片岡 伸介, 濱田 太立, 谷口 理恵子, 川島 希, 西川 英里, 成田 敦, 奥野 友介, 西尾 信博, 小島 大英, 中島 葉子, 柘植 郁哉, 中村 冨美子, 酒井 好美, 伊藤 哲哉, 高橋 義行

    日本小児科学会雑誌   Vol. 126 ( 2 ) page: 281 - 281   2022.2

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  170. New mimic of relapse or regional lymph node metastasis in a cancer survivor: a case of mRNA COVID-19 vaccine-induced lymphadenitis with high FDG uptake

    Tsumura, Y; Asakura, K; Takahashi, I; Akaihata, M; Takahashi, Y; Ishida, Y

    IMMUNOLOGICAL MEDICINE   Vol. 45 ( 1 ) page: 45 - 47   2022.1

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    DOI: 10.1080/25785826.2021.1999786

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  171. Dorsal myelopathy after nelarabine and intrathecal methotrexate therapy

    Sawamura, F; Natsume, J; Nakata, T; Muramatsu, H; Takahashi, Y

    PEDIATRICS INTERNATIONAL   Vol. 64 ( 1 ) page: e15334   2022.1

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    DOI: 10.1111/ped.15334

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  172. Conception by assisted reproductive technology in infants with critical congenital heart disease in Japan.

    Morimoto Y, Go K, Yamamoto H, Fukasawa Y, Nakai M, Morihana E, Yasuda K, Nishikawa H, Ohashi N, Takahashi Y, Kato T

    Reproductive biomedicine online   Vol. 44 ( 1 ) page: 163 - 170   2022.1

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    DOI: 10.1016/j.rbmo.2021.10.005

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  173. Clinical Impact of Melphalan Pharmacokinetics on Transplantation Outcomes in Children Undergoing Hematopoietic Stem Cell Transplantation. International journal

    Ryo Maemura, Manabu Wakamatsu, Kana Matsumoto, Hirotoshi Sakaguchi, Nao Yoshida, Asahito Hama, Taro Yoshida, Shunsuke Miwata, Hironobu Kitazawa, Kotaro Narita, Shinsuke Kataoka, Daisuke Ichikawa, Motoharu Hamada, Rieko Taniguchi, Kyogo Suzuki, Nozomu Kawashima, Eri Nishikawa, Atsushi Narita, Yusuke Okuno, Nobuhiro Nishio, Koji Kato, Seiji Kojima, Kunihiko Morita, Hideki Muramatsu, Yoshiyuki Takahashi

    Cell transplantation   Vol. 31   page: 9636897221143364 - 9636897221143364   2022

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    Melphalan is widely used for hematopoietic stem cell transplantation (HSCT) conditioning. However, the relationship between its pharmacokinetic (PK) and transplantation outcomes in children has not been thoroughly investigated. We prospectively analyzed the relationship between melphalan area under the curve (AUC) and transplantation outcome and examined the development of a predictive model for melphalan clearance in children. This study included 43 children aged 0 to 19 years who underwent HSCT following a melphalan-based conditioning regimen from 2017 to 2021. In univariable analysis, high-melphalan AUC resulted in a significantly lower cumulative incidence of acute graft-versus-host disease and a higher cumulative incidence of thrombotic microangiopathy, although no significant difference was observed in survival. Regression analysis of a randomly selected derivation cohort (n = 21) revealed the following covariate PK model: predicted melphalan clearance (mL/min) = 6.47 × 24-h urinary creatinine excretion rate (CER, g/day) × 24-h creatinine clearance rate (CCR, mL/min) + 92.8. In the validation cohort (n = 22), the measured melphalan clearance values were significantly correlated with those calculated based on the prediction equation (R2 = 0.663). These results indicate that melphalan exposure may be optimized by adjusting the melphalan dose according to CER and CCR.

    DOI: 10.1177/09636897221143364

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  174. Simple and robust methylation test for risk stratification of patients with juvenile myelomonocytic leukemia. International journal

    Hironobu Kitazawa, Yusuke Okuno, Hideki Muramatsu, Kosuke Aoki, Norihiro Murakami, Manabu Wakamatsu, Kyogo Suzuki, Kotaro Narita, Shinsuke Kataoka, Daisuke Ichikawa, Motoharu Hamada, Rieko Taniguchi, Nozomu Kawashima, Eri Nishikawa, Atsushi Narita, Nobuhiro Nishio, Asahito Hama, Mignon L Loh, Elliot Stieglitz, Seiji Kojima, Yoshiyuki Takahashi

    Blood advances   Vol. 5 ( 24 ) page: 5507 - 5518   2021.12

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    Juvenile myelomonocytic leukemia (JMML) is a rare myelodysplastic/myeloproliferative neoplasm that develops during infancy and early childhood. The array-based international consensus definition of DNA methylation has recently classified patients with JMML into the following 3 groups: high (HM), intermediate (IM), and low methylation (LM). To develop a simple and robust methylation clinical test, 137 patients with JMML were analyzed using the Digital Restriction Enzyme Analysis of Methylation (DREAM), which is a next-generation sequencing-based methylation analysis. Unsupervised consensus clustering of the discovery cohort (n = 99) using DREAM data identified HM (HM_DREAM; n = 35) and LM subgroups (LM_DREAM; n = 64). Of the 98 cases that could be compared with the international consensus classification, 90 HM (n = 30) and LM (n = 60) cases had 100% concordance with DREAM clustering results. Of the remaining 8 cases comprising the IM group, 4 were classified as belonging to the HM_DREAM group and 4 to the LM_DREAM group. A machine-learning classifier was successfully constructed using a support vector machine (SVM), which divided the validation cohort (n = 38) into HM (HM_SVM, n = 18) and LM (LM_SVM; n = 20) groups. Patients with the HM_SVM profile had a significantly poorer 5-year overall survival rate than those with the LM_SVM profile. In conclusion, we developed a robust methylation test using DREAM for patients with JMML. This simple and straightforward test can be easily incorporated into diagnosis to generate a methylation classification for patients so they can receive risk-adapted treatment in the context of future clinical trials.

    DOI: 10.1182/bloodadvances.2021005080

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  175. Altered effect of killer immunoglobulin-like receptor-ligand mismatch by graft versus host disease prophylaxis in cord blood transplantation

    Yokoyama, H; Hirayama, M; Takahashi, Y; Uchida, N; Tanaka, M; Onizuka, M; Ozawa, Y; Onai, D; Katsuoka, Y; Wake, A; Sawa, M; Kobayashi, H; Maruyama, Y; Ozeki, K; Kimura, T; Kanda, J; Fukuda, T; Atsuta, Y; Terakura, S; Morishima, S

    BONE MARROW TRANSPLANTATION   Vol. 56 ( 12 ) page: 3059 - 3067   2021.12

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    DOI: 10.1038/s41409-021-01469-6

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  176. Effectiveness of lacosamide in children and young adults previously treated with other sodium channel blockers

    Suzuki, T; Natsume, J; Kumai, S; Maki, Y; Yamamoto, H; Numoto, S; Narahara, S; Kubota, T; Tsuji, T; Kato, T; Yamada, K; Maruyama, K; Okumura, A; Takahashi, Y; Kidokoro, H

    EPILEPSY & BEHAVIOR   Vol. 125   page: 108397   2021.12

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    DOI: 10.1016/j.yebeh.2021.108397

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  177. Prognostic value of the revised International Prognostic Scoring System five-group cytogenetic abnormality classification for the outcome prediction of hematopoietic stem cell transplantation in pediatric myelodysplastic syndrome. International journal

    Shohei Yamamoto, Motohiro Kato, Kenichiro Watanabe, Sae Ishimaru, Daisuke Hasegawa, Maiko Noguchi, Asahito Hama, Maho Sato, Takashi Koike, Fuminori Iwasaki, Hiroshi Yagasaki, Yoshiyuki Takahashi, Yoshiyuki Kosaka, Yoshiko Hashii, Akira Morimoto, Yoshiko Atsuta, Daiichiro Hasegawa, Nao Yoshida

    Bone marrow transplantation   Vol. 56 ( 12 ) page: 3016 - 3023   2021.12

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    Cytogenetic abnormalities are a major risk factor for relapse after hematopoietic stem cell transplantation (HSCT) for myelodysplastic syndrome (MDS). We aimed to evaluate the value of the five-group cytogenetic classification according to the revised International Prognostic Scoring System (R-IPSS) for predicting the outcome after HSCT in pediatric patients with MDS. We retrospectively analyzed the Japanese registration data of 242 pediatric patients with MDS. According to the R-IPSS classification, 112 (45.5%) patients had good, 55 (22.7%) had intermediate, 64 (26.4%) had poor, and 11 (4.6%) had very poor cytogenetics. The 5-year overall survival (5yOS) was 72%, 69%, 59%, and 30% in the good, intermediate, poor, and very poor cytogenetic subgroups (p = 0.026), respectively. The very good, good, and intermediate subgroups were grouped into a "standard" subgroup and reclassified into three subgroups (standard, poor, and very poor). Patients with very poor risk had worse 5yOS (hazard ratio 2.17, 95% confidence interval (CI) 1.02-4.61; p = 0.04) and a much higher 5yCIR (hazard ratio 2.52, 95% CI 1.05-6.04; p = 0.04) than those of patients in the standard group in the multivariate analysis, indicating that very poor risk cytogenetic characteristics independently predicted worse outcome after HSCT in pediatric patients with MDS.

    DOI: 10.1038/s41409-021-01446-z

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  178. [Recommendations (Proposal) for promoting research for overcoming neurological diseases 2020].

    Mochizuki H, Aoki M, Ikenaka K, Inoue H, Iwatsubo T, Ugawa Y, Okazawa H, Ono K, Onodera O, Kitagawa K, Saito Y, Shimohata T, Takahashi R, Toda T, Nakahara J, Matsumoto R, Mizusawa H, Mitsui J, Murayama S, Katsuno M, Future Vision Committee of Japanese Society of Neurology, Aoki Y, Ishiura H, Izumi Y, Koike H, Shimada H, Takahashi Y, Tokuda T, Nakajima H, Hatano T, Misawa S, Watanabe H

    Rinsho shinkeigaku = Clinical neurology   Vol. 61 ( 11 ) page: 709 - 721   2021.11

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    DOI: 10.5692/clinicalneurol.cn-001639

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  179. A Phase I Study of CD19 Chimeric Antigen Receptor-T Cells Generated By the <i>PiggyBac</i> Transposon Vector for Acute Lymphoblastic Leukemia

    Nishio, N; Hanajiri, R; Ishikawa, Y; Murata, M; Taniguchi, R; Hamada, M; Nishikawa, E; Kawashima, N; Narita, A; Muramatsu, H; Takahashi, Y

    BLOOD   Vol. 138   page: 3831 - +   2021.11

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    DOI: 10.1182/blood-2021-150469

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  180. Clinical Features of Immature Leukemias in Children

    Sajiki, D; Yoshida, N; Muramatsu, H; Sakaguchi, K; Maeda, N; Yokoyama, N; Miyajima, Y; Tanaka, M; Takahashi, Y; Hama, A

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  181. Clinical Characteristics and Outcomes of Acute Megakaryoblastic Leukemia with T(1;22)(P13;Q13) in Children

    Hama, A; Nagai, K; Hasegawa, D; Hasegawa, D; Shimada, A; Takahashi, Y; Taga, T; Adachi, S; Tomizawa, D

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  182. Cartilage-Hair Hypoplasia Diagnosed after Optional Screening of Neonates for Severe Combined Immunodeficiency

    Tsumura, Y; Wakamatsu, M; Muramatsu, H; Okuno, Y; Nakajima, Y; Ito, T; Nakamura, T; Sakai, Y; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  183. A Case of FOXN1-SCID Identified by TREC/KREC Newborn Screening Program

    Wakamatsu, M; Kojima, D; Muramatsu, H; Okuno, Y; Kataoka, S; Nakamura, T; Sakai, Y; Nakajima, Y; Ito, T; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  184. A Case of Severe Combined Immunodeficiency found in Newborn Screening for Primary Immunodeficiency in Aichi Prefecture

    Yamashita, D; Wakamatsu, M; Muramatsu, H; Kojima, D; Kataoka, S; Okuno, Y; Nakamura, F; Sakai, Y; Ito, T; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  185. A Case of Fanct without Abnormality in the Peripheral Blood Chromosome Breakage Test

    Nishi, M; Eguchi, H; Muramatsu, H; Takahashi, Y; Matsuo, M

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  186. Hematopoietic Cell Transplantation for Severe Combined Immunodeficiency Patients: a Japanese Retrospective Study. International journal

    Satoshi Miyamoto, Katsutsugu Umeda, Mio Kurata, Akira Nishimura, Masakatsu Yanagimachi, Masataka Ishimura, Maho Sato, Tomonari Shigemura, Motohiro Kato, Yoji Sasahara, Akihiro Iguchi, Takashi Koike, Yoshiyuki Takahashi, Michiko Kajiwara, Masami Inoue, Yoshiko Hashii, Hiromasa Yabe, Koji Kato, Yoshiko Atsuta, Kohsuke Imai, Tomohiro Morio

    Journal of clinical immunology   Vol. 41 ( 8 ) page: 1865 - 1877   2021.11

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    PURPOSE: Hematopoietic cell transplantation (HCT) is a curative therapy for patients with severe combined immunodeficiency (SCID). Here, we conducted a nationwide study to assess the outcome of SCID patients after HCT in Japan. METHODS: A cohort of 181 SCID patients undergoing their first allogeneic HCT in 1974-2016 was studied by using the Japanese national database (Transplant Registry Unified Management Program, TRUMP). RESULTS: The 10-year overall survival (OS) of the patients who received HCT in 2006-2016 was 67%. Umbilical cord blood (UCB) transplantation was performed in 81 patients (45%). The outcomes of HCT from HLA-matched UCB (n = 21) and matched sibling donors (n = 22) were comparable, including 10-year OS (91% vs. 91%), neutrophil recovery (cumulative incidence at 30 days, 89% vs. 100%), and platelet recovery (cumulative incidence at 60 days, 89% vs. 100%). Multivariate analysis of the patients who received HCT in 2006-2016 demonstrated that the following factors were associated with poor OS: bacterial or fungal infection at HCT (hazard ratio (HR): 3.8, P = 0.006), cytomegalovirus infection prior to HCT (HR: 9.4, P = 0.03), ≥ 4 months of age at HCT (HR: 25.5, P = 0.009), and mismatched UCB (HR: 19.8, P = 0.01). CONCLUSION: We showed the potential of HLA-matched UCB as a donor source with higher priority for SCID patients. We also demonstrated that early age at HCT without active infection is critical for a better prognosis, highlighting the importance of newborn screening for SCID.

    DOI: 10.1007/s10875-021-01112-5

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  187. Urinary Biomarkers Predicting Treatment Outcomes in Neuroblastoma

    Atsushi Narita, Akinari Hinoki, Kazuki Yokota, Motoharu Hamada, Shinsuke Kataoka, Nozomu Kawashima, Hideki Muramatsu, Nobuhiro Nishio, Hiroo Uchida, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  188. Using Chemotherapy to Treat Neuroblastoma Stage 4S with Hepatomegaly in a Patient Younger than 2 Months Old

    Masayuki Imaya, Nobuhiro Nishio, Rieko Taniguchi, Shinsuke Kataoka, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Atsushi Narita, Hideki Muramatsu, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  189. Next-Generation Sequencing to Detect Pathogens in Pediatric Febrile Neutropenia: A Single-Center Retrospective Study of 112 Cases

    Kazuhiro Horiba, Yuka Torii, Toshihiko Okumura, Suguru Takeuchi, Takako Suzuki, Jun-ichi Kawada, Hideki Muramatsu, Yoshiyuki Takahashi, Tomoo Ogi, Yoshinori Ito

    Open Forum Infectious Diseases   Vol. 8 ( 11 ) page: ofab223   2021.11

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    <title>Abstract</title>
    <sec>
    <title>Background</title>
    Febrile neutropenia (FN) is a frequent complication in immunocompromised patients. However, causative microorganisms are detected in only 10% of patients. This study aimed to detect the microorganisms that cause FN using next-generation sequencing (NGS) to idenjpgy the genome derived from pathogenic microorganisms in the bloodstream. Here, we implemented a metagenomic approach to comprehensively analyze microorganisms present in clinical samples from patients with FN.


    </sec>
    <sec>
    <title>Methods</title>
    FN is defined as 1) a neutrophil count &amp;lt; 500/µL, and 2) fever ≥ 37.5 °C. Plasma/serum samples of 112 pediatric patients with FN, 10 patients with neutropenia without fever (NE), were sequenced by NGS and analyzed by a metagenomic pipeline PATHDET.


    </sec>
    <sec>
    <title>Results</title>
    The putative pathogens were detected by NGS in 5 of 10 patients with FN with positive for blood culture results, 15 of 87 patients (17%) with negative for blood culture results, and 3 of 8 patients with NE. Several bacteria that were common in the oral, skin, and gut flora were commonly detected in blood samples, suggesting translocation of the human microbiota to the bloodstream in the setting of neutropenia. The cluster analysis of the microbiota in blood samples using NGS demonstrated that the representative bacteria of each cluster was mostly consistent with the pathogens in each patient.


    </sec>
    <sec>
    <title>Conclusions</title>
    NGS technique has a great potential for detecting causative pathogens in patients with FN. Cluster analysis, which extracts characteristic microorganisms from a complex microbial population, may be effective to detect pathogens in minute quantities of microbiota, such as those from the bloodstream.


    </sec>

    DOI: 10.1093/ofid/ofab223

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  190. RE-Treatment with CD19-Targeted CAR-T Cells for Relapse after CD19-CAR-T Cell Therapy

    Nobuhiro Nishio, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Atsushi Narita, Hideki Muramatsu, Kai Wang, Huyong Zheng, Jung-Li Chang, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  191. Prolonged Hospitalization and Late Gastroenterological Complications in Children Undergoing Allogeneic Hematopoietic Stem Cell Transplantation

    Nozomu Kawashima, Shinsuke Kataoka, Motoharu Hamada, Eri Nishikawa, Rieko Taniguchi, Atsushi Narita, Hideki Muramatsu, Nobuhiro Nishio, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  192. Outcomes of KIR-Ligand Incompatible Allogeneic Cord Blood Transplantation for Relapsed Stage 4 Neuroblastoma: A Single Institutional Study

    Shinsuke Kataoka, Nobuhiro Nishio, Manabu Wakamatsu, Rieko Taniguchi, Eri Nishikawa, Motoharu Hamada, Nozomu Kawashima, Atsushi Narita, Hideki Muramatsu, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  193. Strategy of Intensified Therapy for High Risk Pediatric Acute Lymphoblastic Leukemia Based on NGS-MRD-Based Risk Stratification

    Motoharu Hamada, Masayuki Imaya, Yusuke Okuno, Nozomu Kawashima, Rieko Taniguchi, Atsushi Narita, Hideki Muramatsu, Nobuhiro Nishio, Shinji Hasegawa, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  194. Ten Patients with RUNX1 Mutation who were Suspected with FPD-MM in a Pediatric Cohort of Inherited Bone Marrow Failure Syndrome

    Ayako Yamamori, Motoharu Hamada, Hideki Muramatsu, Rieko Taniguchi, Shinsuke Kataoka, Nozomu Kawashima, Atsushi Narita, Nobuhiro Nishio, Yusuke Okuno, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  195. Successful Therapy with Ascites Drainage and Defibrotide for Veno-Occlusive Disease Developed after Bone Marrow Transplantation in Acute Lymphoblastic Leukemia

    Ryo Maemura, Atsushi Narita, Shinsuke Kataoka, Rieko Taniguchi, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Nobuhiro Nishio, Hideki Muramatsu, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 68   2021.11

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  196. がん遺伝子パネル検査でBRAF V600E変異を認めた副腎腫瘍の16歳女性

    佐治木 大知, 村松 秀城, 津村 悠介, 前村 遼, 今屋 雅之, 山森 彩子, 若松 学, 吉田 太郎, 三輪田 俊介, 成田 幸太郎, 市川 大輔, 濱田 太立, 谷口 理恵子, 鈴木 喬悟, 川島 希, 西川 英里, 成田 敦, 西尾 信博, 高橋 義行, 島田 聡子, 榊原 綾子, 下山 芳江, 岩淵 英人

    日本小児血液・がん学会雑誌   Vol. 58 ( 3 ) page: 325 - 326   2021.11

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  197. Hematopoietic stem cell transplantation for infants with high-risk KMT2A gene-rearranged acute lymphoblastic leukemia. International journal

    Takayuki Takachi, Tomoyuki Watanabe, Takako Miyamura, Akiko Moriya Saito, Takao Deguchi, Toshinori Hori, Tomomi Yamada, Shigeru Ohmori, Masami Haba, Yuki Aoki, Sae Ishimaru, Shinya Sasaki, Junjiro Ohshima, Akihiro Iguchi, Yoshiyuki Takahashi, Nobuyuki Hyakuna, Atsushi Manabe, Keizo Horibe, Eiichi Ishii, Katsuyoshi Koh, Daisuke Tomizawa

    Blood advances   Vol. 5 ( 19 ) page: 3891 - 3899   2021.10

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    The role of allogeneic hematopoietic stem cell transplantation (HSCT) for infants with acute lymphoblastic leukemia (ALL) and KMT2A gene rearrangement (KMT2A-r) is controversial in terms of both its efficacy and potential for acute and late toxicities. In Japanese Pediatric Leukemia/Lymphoma Study Group trial MLL-10, by introducing intensive chemotherapy, indication of HSCT was restricted to patients with high-risk (HR) features only (KMT2A-r and either age <180 days or presence of central nervous system leukemia). Of the 56 HR patients, 49 achieved complete remission. Forty-three patients received HSCT in first remission including 38 patients receiving protocol-specified HSCT with conditioning consisting of individualized targeted doses of busulfan, etoposide, and cyclophosphamide. Three-year event-free survival (EFS) of 56.8% (95% confidence interval [CI], 42.4% to 68.8%) and overall survival of 80.2% (95% CI, 67.1% to 88.5%) were accomplished. Univariable analysis showed that Interfant-HR criteria and flow cytometric minimal residual disease (MRD; ≥0.01%), both at the end of induction and at the end of consolidation (EOC), were significantly associated with poorer EFS. In the multivariable analysis, positive MRD at EOC was solely associated with poor EFS (P < .001). Rapid pretransplant MRD clearance and tailored HSCT strategy in the MLL-10 trial resulted in a favorable outcome for infants with HR KMT2A-r ALL. However, considering the high rate of potentially life-threatening toxicities and the risk of late effects, its indication should be further restricted or even eliminated in the future by introducing more effective therapeutic modalities with minimal toxicities. This trial was registered at the University Hospital Medical Information Network Clinical Trials Registry (UMIN-CTR) as #UMIN000004801.

    DOI: 10.1182/bloodadvances.2020004157

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  198. Single-Arm Non-Blinded Multicenter Clinical Trial on T-Cell-Replete Haploidentical Stem Cell Transplantation Using Low-Dose Antithymocyte Globulin for Relapsed and Refractory Pediatric Acute Leukemia.

    Kada A, Kikuta A, Saito AM, Kato K, Iguchi A, Yabe H, Ishida H, Hyakuna N, Takahashi Y, Nagasawa M, Hashii Y, Umeda K, Matsumoto K, Fujisaki H, Yano M, Nakazawa Y, Sano H

    The Kurume medical journal   Vol. 66 ( 3 ) page: 161 - 168   2021.10

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    DOI: 10.2739/kurumemedj.MS663004

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  199. Clinical Images: Extensive multiple organ involvement in VEXAS syndrome

    Takahashi, N; Takeichi, T; Nishida, T; Sato, J; Takahashi, Y; Yamamura, M; Ogi, T; Akiyama, M

    ARTHRITIS & RHEUMATOLOGY   Vol. 73 ( 10 ) page: 1896 - 1897   2021.10

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    DOI: 10.1002/art.41775

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  200. Acquisition of a rare NUP98-BPTF fusion gene associated with recurrence of acute myeloid leukemia. International journal

    Koji Kawaguchi, Shohei Azumi, Yosuke Itakura, Takayuki Takachi, Taemi Ogura, Yasuo Horikoshi, Kyogo Suzuki, Hideki Muramatsu, Asahito Hama, Yoshiyuki Takahashi, Kenichiro Watanabe

    Pediatric blood & cancer   Vol. 68 ( 10 ) page: e29201   2021.10

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    DOI: 10.1002/pbc.29201

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  201. 腹水ドレナージとデフィブロタイドが奏功した急性リンパ性白血病に対する骨髄移植後Veno-Occlusive Disease(Successful Therapy with Ascites Drainage and Defibrotide for Veno-Occlusive Disease Developed After Bone marrow Transplantation in Acute Lymphoblastic Leukemia)

    Maemura Ryo, Narita Atsushi, Kataoka Shinsuke, Taniguchi Rieko, Hamada Motoharu, Nishikawa Eri, Kawashima Nozomu, Nishio Nobuhiro, Muramatsu Hideki, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 246 - 246   2021.10

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  202. 神経芽腫における尿中バイオマーカー(Urinary biomarkers predicting treatment outcomes in neuroblastoma)

    Narita Atsushi, Hinoki Akinari, Yokota Kazuki, Hamada Motoharu, Kataoka Shinsuke, Kawashima Nozomu, Muramatsu Hideki, Nishio Nobuhiro, Uchida Hiroo, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 225 - 225   2021.10

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  203. 小児骨髄不全症候群コホート中のFPD-MMが疑われたRUNX1変異10症例のまとめ(Ten patients with RUNX1 mutation who were suspected with FPD-MM in a pediatric cohort of inherited bone marrow failure syndrome)

    Yamamori Ayako, Hamada Motoharu, Muramatsu Hideki, Taniguchi Rieko, Kataoka Shinsuke, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Okuno Yusuke, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 214 - 214   2021.10

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  204. 化学療法による治療介入を行った生後2ヵ月未満発症の神経芽腫4S(Using chemotherapy to treat neuroblastoma stage 4S with hepatomegaly in a patient younger than 2 months old)

    Imaya Masayuki, Nishio Nobuhiro, Taniguchi Rieko, Kataoka Shinsuke, Hamada Motoharu, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 225 - 225   2021.10

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  205. 単一施設における再発4期神経芽腫に対するKIRリガンド不一致同種臍帯血移植による治療成績(Outcomes of KIR-ligand incompatible allogeneic cord blood transplantation for relapsed stage 4 neuroblastoma: a single institutional study)

    Kataoka Shinsuke, Nishio Nobuhiro, Wakamatsu Manabu, Taniguchi Rieko, Nishikawa Eri, Hamada Motoharu, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 226 - 226   2021.10

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  206. 小児における未分化白血病の臨床的特徴(Clinical features of immature leukemias in children)

    Sajiki Daichi, Yoshida Nao, Muramatsu Hideki, Sakaguchi Kimiyoshi, Maeda Naoko, Yokoyama Norifumi, Miyajima Yuji, Tanaka Makito, Takahashi Yoshiyuki, Hama Asahito

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 185 - 185   2021.10

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  207. 小児同種移植後に長期入院を要する原因と移植後晩期にみられる消化管合併症(Prolonged Hospitalization and Late Gastroenterological Complications in Children Undergoing Allogeneic Hematopoietic Stem Cell Transplantation)

    Kawashima Nozomu, Kataoka Shinsuke, Hamada Motoharu, Nishikawa Eri, Taniguchi Rieko, Narita Atsushi, Muramatsu Hideki, Nishio Nobuhiro, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 242 - 242   2021.10

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  208. NGS-MRDに基づくリスク層別化による高リスクALLに対する治療強化(Strategy of intensified therapy for high risk pediatric acute lymphoblastic leukemia based on NGS-MRD-based risk stratification)

    Hamada Motoharu, Imaya Masayuki, Okuno Yusuke, Kawashima Nozomu, Taniguchi Rieko, Narita Atsushi, Muramatsu Hideki, Nishio Nobuhiro, Hasegawa Shinji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 191 - 191   2021.10

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  209. CD19-CAR-T細胞療法後に再発したB-ALLに対するCD19-CAR-T細胞の再投与(Re-treatment with CD19-targeted CAR-T cells for relapse after CD19-CAR-T cell therapy)

    Nishio Nobuhiro, Hamada Motoharu, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Wang Kai, Zheng Huyong, Chang Jung-Li, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 58 ( 4 ) page: 191 - 191   2021.10

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  210. Usefulness of functional splicing analysis to confirm precise disease pathogenesis in Diamond-Blackfan anemia caused by intronic variants in <i>RPS19</i>

    Takafuji, S; Mori, T; Nishimura, N; Yamamoto, N; Uemura, S; Nozu, K; Terui, K; Toki, T; Ito, E; Muramatsu, H; Takahashi, Y; Matsuo, M; Yamamura, T; Iijima, K

    PEDIATRIC HEMATOLOGY AND ONCOLOGY   Vol. 38 ( 6 ) page: 515 - 527   2021.9

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    DOI: 10.1080/08880018.2021.1887984

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  211. Clinical diagnostic value of telomere length measurement in inherited bone marrow failure syndromes. International journal

    Shunsuke Miwata, Atsushi Narita, Yusuke Okuno, Kyogo Suzuki, Motoharu Hamada, Taro Yoshida, Masayuki Imaya, Ayako Yamamori, Manabu Wakamatsu, Kotaro Narita, Hironobu Kitazawa, Daisuke Ichikawa, Rieko Taniguchi, Nozomu Kawashima, Eri Nishikawa, Nobuhiro Nishio, Seiji Kojima, Hideki Muramatsu, Yoshiyuki Takahashi

    Haematologica   Vol. 106 ( 9 ) page: 2511 - 2515   2021.9

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    DOI: 10.3324/haematol.2021.278334

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  212. Autoantibodies against the plakin family proteins as a novel marker for chronic graft-versus-host disease of the lung. International journal

    Nozomu Kawashima, Eri Nishikawa, Atsunari Tsuchisaka, Takashi Hashimoto, Yusuke Okuno, Motoharu Hamada, Daisuke Ichikawa, Atsushi Narita, Hideki Muramatsu, Nobuhiro Nishio, Seiji Kojima, Yoshinao Muro, Yoshiyuki Takahashi

    Bone marrow transplantation   Vol. 56 ( 9 ) page: 2291 - 2294   2021.9

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    DOI: 10.1038/s41409-021-01335-5

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  213. Echocardiography Monitoring of Pulmonary Hypertension after Pediatric Hematopoietic Stem Cell Transplantation: Pediatric Pulmonary Arterial Hypertension and Pulmonary Veno-Occlusive Disease after Hematopoietic Stem Cell Transplantation. International journal

    Nozomu Kawashima, Yoshie Fukasawa, Eri Nishikawa, Keiko Ohta-Ogo, Hatsue Ishibashi-Ueda, Motoharu Hamada, Daisuke Ichikawa, Atsushi Narita, Yusuke Okuno, Hideki Muramatsu, Nobuhiro Nishio, Seiji Kojima, Taichi Kato, Yoshiyuki Takahashi

    Transplantation and cellular therapy   Vol. 27 ( 9 ) page: 786.e1 - 786.e8   2021.9

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    Pulmonary hypertension (PH) is associated with high morbidity in children undergoing hematopoietic stem cell transplantation (HSCT). However, owing to the lack of sequential echocardiography, the nature of the condition is not fully understood. This study was conducted to investigate whether routine echocardiography performed after HSCT could detect patients with PH at an earlier stage and elucidate the role of intervention using tadalafil. The study population comprised 93 consecutive children age <18 years who underwent a total of 109 HSCTs. All patients underwent routine transthoracic echocardiography during HSCT. Four children (4%) with a median age of 4 years (range, 0.7 to 6 years) were found to have PH, and their median tricuspid regurgitation peak velocity (TRV) was 4.1 m/s (range, 3.5 to 4.2 m/s). PH was diagnosed at a median of 52 days (range, 21 to 118 days) after HSCT. Three of them were diagnosed with neuroblastoma, and 1 was diagnosed with infantile leukemia. One patient developed PH after autologous HSCT, and 3 received killer immunoglobulin-like receptor ligand-mismatched cord blood. Busulfan was used for conditioning in all patients, and the proportion of patients receiving this medication was significantly higher in the PH group compared with the non-PH group (100% versus 30%; P = .011). Three of the 4 patients had a durable response (TRV ≤2.8 m/s) at a median of 46 days (range, 14 to 79 days) after starting treatment with tadalafil. No patient experienced exacerbation of PH, and treatment was completed at median of 96 days (range, 46 to 212 days). Our data suggest that routine echocardiography monitoring after HSCT should be considered in children receiving busulfan, although the precise follow-up timing needs further study. In addition, safe and effective administration of tadalafil must be ensured by close monitoring.

    DOI: 10.1016/j.jtct.2021.05.017

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  214. Effect of high-dose chemotherapy plus stem cell rescue on the survival of patients with neuroblastoma modified by MYCN gene gain/amplification and remission status: a nationwide registration study in Japan. International journal

    Yuya Saito, Mitsuyoshi Urashima, Yoshiyuki Takahashi, Atsushi Ogawa, Chikako Kiyotani, Yuki Yuza, Katsuyoshi Koh, Kenichiro Watanabe, Yoshiyuki Kosaka, Hiroaki Goto, Atsushi Kikuta, Keiko Okada, Yuhki Koga, Junya Fujimura, Masami Inoue, Atsushi Sato, Yoshiko Atsuta, Kimikazu Matsumoto

    Bone marrow transplantation   Vol. 56 ( 9 ) page: 2173 - 2182   2021.9

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    In high-risk neuroblastoma, the presence of an MYCN gain/amplification (MYCN-GA) is not always a risk factor of cancer-specific death. We herein examined the effect modification of high-dose chemotherapy with autologous hematopoietic stem cell rescue (HDC-autoSCR) in terms of the interaction between MYCN status and remission status (complete remission or very good partial remission [CR/VGPR] vs. partial remission or less [≤PR]). The present study recruited patient data from 1992 to 2017 in the Japan Society of Hematopoietic Cell Transplantation's national registry. The MYCN status was known in 586 of 950 patients with a single course of HDC-autoSCR. Cumulative hazard curves for neuroblastoma-specific death showed that a subgroup with MYCN-GA and ≤PR had a significantly poorer prognosis than three other subgroups, namely, the MYCN-NGA/ ≤ PR, MYCN-NGA/CR/VGPR, and MYCN-GA/CR/VGPR subgroups even after adjusting for non-infants and stage IV disease (hazard ratio: 2.79; 95% confidence interval: 1.91-4.09; P < 0.001). The interaction between MYCN-GA and ≤PR was significant (pinteraction = 0.006). Hence, the patients with MYCN-GA with non-remission status at HDC-autoSCR had a significantly poorer prognosis than the other subgroups, suggesting that HDC-autoSCR may be effective in patients with CR/VGPR regardless of MYCN gene status and in patients with MYCN-NGA regardless of remission status.

    DOI: 10.1038/s41409-021-01303-z

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  215. 急性リンパ性白血病に対するpiggyBacトランスポゾン法によるCD19 CAR-T療法の臨床第I相試験(Phase I study of piggyBac transposon mediated CD19: CAR-T therapy for acute lymphoblastic leukemia)

    西尾 信博, 葉名尻 良, 石川 裕一, 寺倉 精太郎, 西田 徹也, 村田 誠, 濱田 太立, 西川 英里, 川島 希, 奥野 友介, 成田 敦, 村松 秀城, 高橋 義行

    日本血液学会学術集会   Vol. 83回   page: OS2 - 4   2021.9

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  216. Revesz syndrome with severe pancytopenia and bilateral retinal detachment: A case report(和訳中)

    山森 彩子, 津村 悠介, 前村 遼, 今屋 雅之, 若松 学, 三輪田 俊介, 吉田 太郎, 成田 幸太郎, 谷口 理恵子, 濱田 太立, 鈴木 喬悟, 西川 英里, 川島 希, 成田 敦, 村松 秀城, 奥野 友介, 西尾 信博, 小島 勢二, 高橋 義行

    日本血液学会学術集会   Vol. 83回   page: PS - 4   2021.9

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  217. Prospective validation of the provisional entity of RCC proposed by the WHO(和訳中)

    濱 麻人, 真部 淳, 長谷川 大輔, 野沢 和江, 成田 敦, 村松 秀城, 高橋 義行, 渡邉 健一郎, 小原 明, 伊藤 雅文, 小島 勢二

    日本血液学会学術集会   Vol. 83回   page: OS1 - 2   2021.9

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  218. HLA one-allele mismatched unrelated BMT using antithymocyte globulin in Japanese pediatric patients(和訳中)

    濱田 太立, 村松 秀城, 吉田 太郎, 今屋 雅之, 山森 彩子, 三輪田 俊介, 成田 幸太郎, 鈴木 喬悟, 川島 希, 谷口 理恵子, 西川 英里, 成田 敦, 西尾 信博, 高橋 義行

    日本血液学会学術集会   Vol. 83回   page: OS1 - 4   2021.9

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  219. Phase I study of piggyBac transposon mediated CD19: CAR-T therapy for acute lymphoblastic leukemia(和訳中)

    西尾 信博, 葉名尻 良, 石川 裕一, 寺倉 精太郎, 西田 徹也, 村田 誠, 濱田 太立, 西川 英里, 川島 希, 奥野 友介, 成田 敦, 村松 秀城, 高橋 義行

    日本血液学会学術集会   Vol. 83回   page: OS2 - 4   2021.9

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  220. Blinatumomab in children with relapsed/refractory B-cell precursor acute lymphoblastic leukemia(和訳中)

    成田 敦, 佐治木 大知, 津村 悠介, 前村 遼, 今屋 雅之, 山森 彩子, 若松 学, 谷口 理恵子, 濱田 太立, 片岡 伸介, 西川 英里, 川島 希, 奥野 友介, 村松 秀城, 西尾 信博, 高橋 義行

    日本血液学会学術集会   Vol. 83回   page: OS1 - 2   2021.9

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  221. A mutation in a proteasome subunit depletes its activities leading to excessive interferon signaling(和訳中)

    川島 希, 片岡 伸介, 村松 秀城, 大西 秀典, 奥野 友介, 今屋 雅之, 山森 彩子, 若松 学, 濱田 太立, 西川 英里, 成田 敦, 西尾 信博, 小島 勢二, 高橋 義行

    日本血液学会学術集会   Vol. 83回   page: OS1 - 5   2021.9

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  222. Relationship between plasma rabbit anti-thymocyte globulin concentration and immunosuppressive therapy response in patients with severe aplastic anemia. International journal

    Atsushi Narita, Hideki Muramatsu, Daisuke Ichikawa, Motoharu Hamada, Eri Nishikawa, Kyogo Suzuki, Nozomu Kawashima, Yusuke Okuno, Nobuhiro Nishio, Asahito Hama, Hirohito Yamazaki, Shinji Nakao, Seiji Kojima, Yoshiyuki Takahashi

    European journal of haematology   Vol. 107 ( 2 ) page: 255 - 264   2021.8

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    OBJECTIVES: Patients with acquired aplastic anemia (AA) without HLA-matched sibling donors or aged >40 years receive immunosuppressive therapy (IST) with anti-thymocyte globulin (ATG). We investigated the relationship between plasma rabbit ATG (r-ATG) concentration and IST response. METHODS: From May 2012 to October 2017, 81 patients with severe AA who required initial IST were included. A 1:1 block randomization was employed for 2.5 and 3.5 mg/kg doses of r-ATG. RESULTS: No significant difference in response rates was observed between the 2.5 and 3.5 mg/kg groups (63% vs. 58%, P = .894). Median r-ATG concentrations on days 14 and 28 after IST were 15.2 (0.0-97.7) and 1.8 (0.0-74.9 µg/mL), respectively. According to r-ATG concentration, response rates were significantly higher in the group with higher r-ATG concentration than in those with lower r-ATG concentration (day 14, 88% vs. 52%; P = .006 and day 28, 79% vs. 46%; P = .005). In multivariate analysis, higher r-ATG concentrations at day 28 were independent predictors of favorable response to IST at 6 months (odds ratio, 0.29; 95% confidence interval, 0.09-0.93; P = .037). CONCLUSIONS: The present data indicate that higher r-ATG concentration at day 28 resulted in improved IST response.

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  223. Repetitive sleep starts: An important differential diagnosis of infantile spasms. International journal

    Yuki Maki, Hiroyuki Kidokoro, Akihisa Okumura, Hiroyuki Yamamoto, Tomohiko Nakata, Tatsuya Fukasawa, Tetsuo Kubota, Masahiro Kawaguchi, Takeshi Suzuki, Masaharu Tanaka, Yu Okai, Yoko Sakaguchi, Atsuko Ohno, Tamiko Negoro, Yoshiyuki Takahashi, Jun Natsume

    Epilepsy & behavior : E&B   Vol. 121 ( Pt A ) page: 108075 - 108075   2021.8

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    OBJECTIVE: Repetitive sleep starts (RSS) are clusters of nonepileptic, spasm-like movements occurring during sleep onset. However, their characteristics have yet to be defined. We conducted a clinicoelectroencephalographic study of children with RSS to clarify their detailed characteristics. METHODS: To differentiate starts from epileptic spasms, we recruited children with brief "crescendo-decrescendo" muscle contractions that simultaneously involved the limbs and trunk without electroencephalogram changes, and that fulfilled the following criteria: (1) repeated occurrence (five or more) and (2) manifestation during sleep stage N1-N2. A total of nine children met these criteria. Their clinical information and video-electroencephalogram data were analyzed retrospectively. RESULTS: The background conditions observed at onset of RSS were perinatal hypoxic-ischemic encephalopathy (n = 4), West syndrome of unknown etiology (n = 1), and traumatic brain injury (n = 1). The age at onset of RSS, the number of starts in a given RSS cluster, the interval between starts, and the duration of surface electromyogram activity were between 3 and 46 months, 5 and 547, <1 and 60 s, and 0.3 and 5.4 s, respectively. None of the median value of these parameters differed between children with and without corticospinal tract injury. During the median follow-up period of 33 months, RSS disappeared spontaneously in five. CONCLUSION: This is the largest case series of RSS clarifying their clinicoelectroencephalographic characteristics reported to date. To avoid unnecessary antiepileptic therapies, clinicians should be aware of RSS and distinguish it from other disorders involving involuntary movements or seizures, especially epileptic spasms.

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  224. Successful treatment of a novel type I interferonopathy due to a de novo PSMB9 gene mutation with a Janus kinase inhibitor

    Shinsuke Kataoka, Nozomu Kawashima, Yusuke Okuno, Hideki Muramatsu, Shunsuke Miwata, Kotaro Narita, Motoharu Hamada, Norihiro Murakami, Rieko Taniguchi, Daisuke Ichikawa, Hironobu Kitazawa, Kyogo Suzuki, Eri Nishikawa, Atsushi Narita, Nobuhiro Nishio, Hidenori Yamamoto, Yoshie Fukasawa, Taichi Kato, Hiroyuki Yamamoto, Jun Natsume, Seiji Kojima, Ichizo Nishino, Takeshi Taketani, Hidenori Ohnishi, Yoshiyuki Takahashi

    Journal of Allergy and Clinical Immunology   Vol. 148 ( 2 ) page: 639 - 644   2021.8

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    DOI: 10.1016/j.jaci.2021.03.010

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  225. Intravenously delivered multilineage-differentiating stress enduring cells dampen excessive glutamate metabolism and microglial activation in experimental perinatal hypoxic ischemic encephalopathy

    Suzuki, T; Sato, Y; Kushida, Y; Tsuji, M; Wakao, S; Ueda, K; Imai, K; Iitani, Y; Shimizu, S; Hida, H; Temma, T; Saito, S; Iida, H; Mizuno, M; Takahashi, Y; Dezawa, M; Borlongan, CV; Hayakawa, M

    JOURNAL OF CEREBRAL BLOOD FLOW AND METABOLISM   Vol. 41 ( 7 ) page: 1707 - 1720   2021.7

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    DOI: 10.1177/0271678X20972656

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  226. 小児におけるcord colitis syndromeの臨床的・病理学的特徴

    前村 遼, 成田 敦, 濱田 太立, 鈴木 喬悟, 西川 英里, 川島 希, 奥野 友介, 西尾 信博, 村松 秀城, 伊藤 雅文, 高橋 義行

    臨床血液   Vol. 62 ( 7 ) page: 863 - 863   2021.7

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  227. Integrated diagnosis based on transcriptome analysis in suspected pediatric sarcomas. International journal

    Daisuke Ichikawa, Kyoko Yamashita, Yusuke Okuno, Hideki Muramatsu, Norihiro Murakami, Kyogo Suzuki, Daiei Kojima, Shinsuke Kataoka, Motoharu Hamada, Rieko Taniguchi, Eri Nishikawa, Nozomu Kawashima, Atsushi Narita, Nobuhiro Nishio, Asahito Hama, Kenji Kasai, Seiji Mizuno, Yoshie Shimoyama, Masato Nakaguro, Hajime Okita, Seiji Kojima, Atsuko Nakazawa, Yoshiyuki Takahashi

    NPJ genomic medicine   Vol. 6 ( 1 ) page: 49 - 49   2021.6

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    Pediatric solid tumors are a heterogeneous group of neoplasms with over 100 subtypes. Clinical and histopathological diagnosis remains challenging due to the overlapping morphological and immunohistochemical findings and the presence of atypical cases. To evaluate the potential utility of including RNA-sequencing (RNA-seq) in the diagnostic process, we performed RNA-seq in 47 patients with suspected pediatric sarcomas. Histopathologists specialized in pediatric cancer re-evaluated pathological specimens to reach a consensus diagnosis; 42 patients were diagnosed with known subtypes of solid tumors whereas 5 patients were diagnosed with undifferentiated sarcoma. RNA-seq analysis confirmed and refined consensus diagnoses and further identified diagnostic genetic variants in four of the five patients with undifferentiated sarcoma. Genetic lesions were detected in 23 patients, including the novel SMARCA4-THOP1 fusion gene and 22 conventional or recently reported genetic events. Unsupervised clustering analysis of the RNA-seq data identified a distinct cluster defined by the overexpression of rhabdomyosarcoma-associated genes including MYOG and CHRNG. These findings suggest that RNA-seq-based genetic analysis may aid in the diagnosis of suspected pediatric sarcomas, which would be useful for the development of stratified treatment strategies.

    DOI: 10.1038/s41525-021-00210-y

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  228. Age estimates from brain magnetic resonance images of children younger than two years of age using deep learning

    Kawaguchi, M; Kidokoro, H; Ito, R; Shiraki, A; Suzuki, T; Maki, Y; Tanaka, M; Sakaguchi, Y; Yamamoto, H; Takahashi, Y; Naganawa, S; Natsume, J

    MAGNETIC RESONANCE IMAGING   Vol. 79   page: 38 - 44   2021.6

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    DOI: 10.1016/j.mri.2021.03.004

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  229. A Phase 2 Open-label, Single-arm, Multicenter Study of Ruxolitinib Added to Corticosteroids in Pediatric Subjects with Moderate/Severe Chronic Gvhd After Allogeneic Stem Cell Transplantation (REACH5)

    Wall, D; Koh, K; Bhat, S; Takahashi, Y; Zhang, A; Rosko, C; Smith, Y; Stefanelli, T; Diaz-de-Heredia, C; Locatelli, F

    BONE MARROW TRANSPLANTATION   Vol. 56 ( SUPPL 1 ) page: 228 - 229   2021.6

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  230. 診断および治療に難渋している、脳を含む全身性多発転移腫瘤を有する先天性肉腫の乳児

    今屋 雅之, 川島 希, 山森 彩子, 若松 学, 吉田 太郎, 三輪田 俊介, 成田 幸太郎, 北澤 宏展, 谷口 理恵子, 市川 大輔, 濱田 太立, 西川 英里, 成田 敦, 奥野 友介, 村松 秀城, 西尾 信博, 小島 勢二, 高橋 義行, 中村 栄男, 孝橋 賢一, 大喜多 肇

    日本小児血液・がん学会雑誌   Vol. 58 ( 1 ) page: 60 - 60   2021.6

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  231. がん遺伝子パネル検査でGOPC-ROS1 fusionを認めた中枢神経胎児性腫瘍の0歳児症例

    夏目 敦至, 栗本 路弘, 芝 良樹, 青木 恒介, 若林 俊彦, 川島 希, 谷口 理恵子, 村松 秀城, 高橋 義行, 高野 桂, 榊原 綾子, 下山 芳江, 中村 栄男, 森田 佐知, 安藤 雄一, 平戸 純子

    日本小児血液・がん学会雑誌   Vol. 58 ( 1 ) page: 61 - 61   2021.6

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  232. Urinary biomarkers for monitoring treatment response in neuroblastoma patients.

    Amano, H; Hinoki, A; Uchida, H; Yokota, K; Ishigaki, T; Sakairi, M; Abe, M; Takahashi, Y; Narita, A; Tainaka, T; Shirota, C; Sumida, W; Makita, S; Takimoto, A; Kano, Y; Yasui, A; Okamoto, M; Nakagawa, Y

    JOURNAL OF CLINICAL ONCOLOGY   Vol. 39 ( 15 )   2021.5

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    DOI: 10.1200/JCO.2021.39.15_suppl.e22008

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  233. Whole genome sequencing of 45 Japanese patients with intellectual disability

    Abe-Hatano, C; Iida, A; Kosugi, S; Momozawa, Y; Terao, C; Ishikawa, K; Okubo, M; Hachiya, Y; Nishida, H; Nakamura, K; Miyata, R; Murakami, C; Takahashi, K; Hoshino, K; Sakamoto, H; Ohta, S; Kubota, M; Takeshita, E; Ishiyama, A; Nakagawa, E; Sasaki, M; Kato, M; Matsumoto, N; Kamatani, Y; Kubo, M; Takahashi, Y; Natsume, J; Inoue, K; Goto, YI

    AMERICAN JOURNAL OF MEDICAL GENETICS PART A   Vol. 185 ( 5 ) page: 1468 - 1480   2021.5

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    DOI: 10.1002/ajmg.a.62138

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  234. Reduced-intensity conditioning is effective for hematopoietic stem cell transplantation in young pediatric patients with Diamond–Blackfan anemia

    Shun Koyamaishi, Takuya Kamio, Akie Kobayashi, Tomohiko Sato, Ko Kudo, Shinya Sasaki, Rika Kanezaki, Daiichiro Hasegawa, Hideki Muramatsu, Yoshiyuki Takahashi, Yoji Sasahara, Hidefumi Hiramatsu, Harumi Kakuda, Miyuki Tanaka, Masataka Ishimura, Masanori Nishi, Akira Ishiguro, Hiromasa Yabe, Takeo Sarashina, Masaki Yamamoto, Yuki Yuza, Nobuyuki Hyakuna, Kenichi Yoshida, Hitoshi Kanno, Shouichi Ohga, Akira Ohara, Seiji Kojima, Satoru Miyano, Seishi Ogawa, Tsutomu Toki, Kiminori Terui, Etsuro Ito

    Bone Marrow Transplantation   Vol. 56 ( 5 ) page: 1013 - 1020   2021.5

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    Allogeneic hematopoietic stem cell transplantation (HSCT) is the only curative therapy for the hematologic manifestations of Diamond–Blackfan anemia (DBA). However, data regarding the optimal conditioning regimen for DBA patients are limited. We retrospectively compared the outcomes of DBA patients who underwent HSCT using either myeloablative conditioning (MAC) or reduced-intensity conditioning (RIC) regimens. The patients belonged to a cohort treated at our hospitals between 2000 and 2018. HSCT was performed in 27 of 165 patients (16.4%). The median age at the time of HSCT was 3.6 years. Stem cell sources included bone marrow for 25 patients (HLA-matched sibling donors, n = 5; HLA-mismatched related donors, n = 2; HLA-matched/mismatched unrelated donors, n = 18) or cord blood for 2 patients. MAC or RIC regimens were used in 12 and 15 patients, respectively. Engraftment was successful in all 27 patients who underwent HSCT. Three patients who underwent HSCT using MAC regimens developed sinusoidal obstruction syndrome. The 3-year overall survival (OS) and failure-free survival rates (FFS) post-transplantations were 95.2% and 88.4%, respectively, with no significant differences between MAC and RIC regimens. Our data suggest that HSCTs using RIC regimens are effective and obtain engraftment with excellent OS and FFS for young DBA patients.

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  235. Correction: Reduced-intensity conditioning is effective for hematopoietic stem cell transplantation in young pediatric patients with Diamond-Blackfan anemia (Bone Marrow Transplantation, (2021), 56, 5, (1013-1020), 10.1038/s41409-020-01056-1)

    Shun Koyamaishi, Takuya Kamio, Akie Kobayashi, Tomohiko Sato, Ko Kudo, Shinya Sasaki, Rika Kanezaki, Daiichiro Hasegawa, Hideki Muramatsu, Yoshiyuki Takahashi, Yoji Sasahara, Hidefumi Hiramatsu, Harumi Kakuda, Miyuki Tanaka, Masataka Ishimura, Masanori Nishi, Akira Ishiguro, Hiromasa Yabe, Takeo Sarashina, Masaki Yamamoto, Yuki Yuza, Nobuyuki Hyakuna, Kenichi Yoshida, Hitoshi Kanno, Shouichi Ohga, Akira Ohara, Seiji Kojima, Satoru Miyano, Seishi Ogawa, Tsutomu Toki, Kiminori Terui, Etsuro Ito

    Bone Marrow Transplantation   Vol. 56 ( 5 ) page: 1218 - 1219   2021.5

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    In the original version of this Article, in Table 1 the Year of HST for cases 3, 4, 15, and 25 was not shown. They now appear in Table 1 in the PDF and HTML versions of the Article.

    DOI: 10.1038/s41409-020-01076-x

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  236. Temporal dynamics of the plasma microbiome in recipients at early post-liver transplantation: a retrospective study. International journal

    Toshihiko Okumura, Kazuhiro Horiba, Hideya Kamei, Suguru Takeuchi, Takako Suzuki, Yuka Torii, Jun-Ichi Kawada, Yoshiyuki Takahashi, Yasuhiro Ogura, Tomoo Ogi, Yoshinori Ito

    BMC microbiology   Vol. 21 ( 1 ) page: 104 - 104   2021.4

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    BACKGROUND: Immunosuppression during liver transplantation (LT) enables the prevention and treatment of organ rejection but poses a risk for severe infectious diseases. Immune modulation and antimicrobials affect the plasma microbiome. Thus, determining the impact of immunosuppression on the microbiome may be important to understand immunocompetence, elucidate the source of infection, and predict the risk of infection in LT recipients. We characterized the plasma microbiome of LT recipients at early post-LT and assessed the association between the microbiome and clinical events. RESULTS: In this study, 51 patients who received LT at Nagoya University Hospital from 2016 to 2018 were enrolled. Plasma samples were retrospectively collected at the following time points: 1) within a week after LT; 2) 4 ± 1 weeks after LT; 3) 8 ± 1 weeks after LT; and 4) within 2 days after a positive blood culture. A total of 111 plasma samples were analyzed using shotgun next-generation sequencing (NGS) with the PATHDET pipeline. Relative abundance of Anelloviridae, Nocardiaceae, Microbacteriaceae, and Enterobacteriaceae significantly changed during the postoperative period. Microbiome diversity was higher within a week after LT than that at 8 weeks after LT. Antimicrobials were significantly associated with the microbiome of LT recipients. In addition, the proportion of Enterobacteriaceae was significantly increased and the plasma microbiome diversity was significantly lower in patients with acute cellular rejection (ACR) than non-ACR patients. Sequencing reads of bacteria isolated from blood cultures were predominantly identified by NGS in 8 of 16 samples, and human herpesvirus 6 was detected as a causative pathogen in one recipient with severe clinical condition. CONCLUSIONS: The metagenomic NGS technique has great potential in revealing the plasma microbiome and is useful as a comprehensive diagnostic procedure in clinical settings. Temporal dynamics of specific microorganisms may be used as indirect markers for the determination of immunocompetence and ACR in LT recipients.

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  237. Hippocampal Atrophy in Pediatric Transplant Recipients with Human Herpesvirus 6B. International journal

    Misa Miyake, Yoshiki Kawamura, Naoko Ishihara, Shigetaka Suzuki, Hiroki Miura, Yoko Sakaguchi, Masaharu Tanaka, Yoshiyuki Takahashi, Seiji Kojima, Hiroshi Toyama, Jun Natsume, Tetsushi Yoshikawa

    Microorganisms   Vol. 9 ( 4 )   2021.4

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    The aim of this study was to determine whether human herpesvirus 6B (HHV-6B) infection can impair the hippocampus in pediatric hematopoietic stem cell transplant (HSCT) recipients. Study subjects were pediatric HSCT recipients monitored for HHV-6B infection who underwent brain MRI before and after transplantation. Volumetric analysis of the hippocampus was performed. Of the 107 patients that received HSCT at Nagoya University Hospital Between July 2008 and April 2014, 20 were eligible for volumetric analysis. Eight patients had HHV-6B infection, of whom two had encephalopathy at the time of HHV-6B infection. None of the 12 patients without HHV-6B infection had encephalopathy. The median ratio of the right hippocampal volume from before to after transplantation was 0.93 in patients with HHV-6B infection and 1.02 in without HHV-6B infection (p = 0.007). The median ratio of the left hippocampal volume ratio in patients with and without HHV-6B infection was 0.92 and 1.00, respectively (p = 0.003). Among the eight patients with HHV-6B infection, four had a marked reduction in hippocampal volume (volume ratio < 0.90). Only one of these patients had neurological symptoms at the time of HHV-6B infection. The reduction in the hippocampal volume ratio was higher in pediatric HSCT recipients with HHV-6B infection than those without viral infection. Neurological follow-up may be required for pediatric HSCT recipients with HHV-6B infection.

    DOI: 10.3390/microorganisms9040776

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  238. Phase I clinical trial of intra-bone marrow cotransplantation of mesenchymal stem cells in cord blood transplantation

    Goto, T; Murata, M; Nishida, T; Terakura, S; Kamoshita, S; Ishikawa, Y; Ushijima, Y; Adachi, Y; Suzuki, S; Kato, K; Hirakawa, A; Nishiwaki, S; Nishio, N; Takahashi, Y; Kodera, Y; Matsushita, T; Kiyoi, H

    STEM CELLS TRANSLATIONAL MEDICINE   Vol. 10 ( 4 ) page: 542 - 553   2021.4

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    DOI: 10.1002/sctm.20-0381

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  239. Phosphorylated proteome analysis of a novel germline ABL1 mutation causing an autosomal dominant syndrome with ventricular septal defect. International journal

    Hidenori Yamamoto, Satoshi Hayano, Yusuke Okuno, Atsuto Onoda, Kohji Kato, Noriko Nagai, Yoshie Fukasawa, Shinji Saitoh, Yoshiyuki Takahashi, Taichi Kato

    International journal of cardiology   Vol. 326   page: 81 - 87   2021.3

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    BACKGROUND: A gain-of-function mutation in germline ABL1 causes a syndrome including congenital heart defects. However, the molecular mechanisms of this syndrome remain unknown. In this study, we found a novel ABL1 mutation in a Japanese family with ventricular septal defect, finger contracture, skin abnormalities and failure to thrive, and the molecular mechanisms of these phenotypes were investigated. METHODS AND RESULTS: Whole-exome sequencing on several family members revealed a novel mutation (c.1522A > C, p.I508L) in the tyrosine kinase domain of ABL1, and complete co-segregation with clinical presentations was confirmed in all members. Wild-type and mutant ABL1 were transfected into human embryonic kidney 293 cells for functional analysis. Western blotting confirmed that tyrosine phosphorylation in STAT5, a substrate of ABL1, was enhanced, and the novel mutation was proved to be a gain-of-function mutation. Since this novel mutation in ABL1 enhances tyrosine kinase activity, phosphorylated proteome analysis was used to elucidate the molecular pathology. The proteome analysis showed that phosphorylation in proteins such as UFD1, AXIN1, ATRX, which may be involved in the phenotypes, was enhanced in the mutant group. CONCLUSIONS: The onset of congenital heart defects associated with this syndrome appears to involve a mechanism caused by UFD1 common to 22q.11.2 deletion syndrome. On the other hand, AXIN1 and ATRX may be important in elucidating the mechanisms of other phenotypes, such as finger contracture and failure to thrive. Verification of these hypotheses would lead to further understanding of the pathophysiology and the development of treatment methods.

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  240. Initial treatment of seizures in children in an emergency department in rural Japan. International journal

    Anna Shiraki, Masahiro Yasui, Hiroyuki Kidokoro, Shinji Kido, Hideo Ando, Yoshiyuki Takahashi, Jun Natsume

    Brain & development   Vol. 43 ( 2 ) page: 288 - 293   2021.2

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    OBJECTIVE: Although the initial treatment of childhood seizures is important, treatment within an appropriate time window is often difficult in resource-limited areas. This study examined childhood seizure treatment in a rural area in Japan. METHODS: We retrospectively investigated children presenting to Nakatsugawa Municipal General Hospital emergency department between 2015 and 2018. From the hospital database, we identified children who were diagnosed with seizures, epilepsy, or acute infectious encephalitis/encephalopathy or were given benzodiazepines. We considered etiology, seizure duration, and treatment according to the specialties of the doctors providing initial care. RESULTS: We extracted 236 seizure events: 40 initially treated by pediatricians, 16 by a mobile doctor team, and 180 by other doctors. Twenty patients had continuous seizures for longer than 5 min on admission. Two were treated by pediatricians at presentation; it took 4 and 7 min after arrival to stop the seizures. Four were treated by a mobile team, and 14 by other doctors; the median response times were 11.5 (range 3-47) and 19 (range 5-60) min, respectively. All patients treated by pediatricians or mobile doctor teams received intravenous or intramuscular diazepam, whereas 50% of those treated by other doctors initially received diazepam suppositories. In three of the 20 events, establishing intravenous access was difficult. SIGNIFICANCE: In rural Japan, many children with seizures are initially treated by doctors other than pediatricians or emergency physicians, and they require a longer time to achieve seizure cessation. Non-intravenous benzodiazepine formulas, which have not yet been approved in Japan, would be helpful.

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  241. Combination of tumor necrosis factor-α and epidermal growth factor induces the adrenergic-to-mesenchymal transdifferentiation in SH-SY5Y neuroblastoma cells. International journal

    Yue Huang, Shoma Tsubota, Nobuhiro Nishio, Yoshiyuki Takahashi, Kenji Kadomatsu

    Cancer science   Vol. 112 ( 2 ) page: 715 - 724   2021.2

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    Neuroblastoma, a type of cancer that is common in children, is composed of two genetically clonal but epigenetically distinct cell types: mesenchymal (MES) and adrenergic (ADRN) types, controlled by super-enhancer-associated lineage-specific transcription factor networks. Mesenchymal-type cells are more migratory, resistant to chemotherapy, and prevalent in relapse tumors. Importantly, both cell types spontaneously transdifferentiate into one another, and this interconversion can be induced by genetic manipulations. However, the mechanisms of their spontaneous transdifferentiation and extracellular factors inducing this phenomenon have not yet been elucidated. Using a unique approach involving gene set enrichment analysis, we selected six ADRN and 10 MES candidate factors, possibly inducing ADRN and MES phenotypes, respectively. Treatment with a combination of 10 MES factors clearly induced the MES gene expression profile in ADRN-type SH-SY5Y neuroblastoma cells. Considering the effects on gene expression profile, migration ability, and chemoresistance, a combination of tumor necrosis factor alpha (TNF-α) and epidermal growth factor (EGF) was sufficient to synergistically induce the ADRN-to-MES transdifferentiation in SH-SY5Y cells. In addition, human neuroblastoma cohort analysis revealed that the expression of TNF and EGF receptors was strongly associated with MES gene expression signatures, supporting their important roles in transdifferentiation in vivo. Collectively, we propose a mechanism of neuroblastoma transdifferentiation induced by extracellular growth factors, which can be controlled in clinical situations, providing a new therapeutic possibility.

    DOI: 10.1111/cas.14760

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  242. Shuffling babies and autism spectrum disorder. International journal

    Yu Okai, Tomohiko Nakata, Kiyokuni Miura, Atsuko Ohno, Rie Wakako, Osamu Takahashi, Yuki Maki, Masaharu Tanaka, Yoko Sakaguchi, Yuji Ito, Hiroyuki Yamamoto, Hiroyuki Kidokoro, Yoshiyuki Takahashi, Jun Natsume

    Brain & development   Vol. 43 ( 2 ) page: 181 - 185   2021.2

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    BACKGROUND AND PURPOSE: Bottom shuffling is a locomotion strategy that precedes independent walking in some infants. Shuffling babies are generally considered to have favorable outcomes. The aim of the present study was to reveal clinical features and neurodevelopmental outcomes of shuffling babies who visited a child developmental center. METHODS: We studied 48 shuffling babies who visited Toyota Municipal Child Development Center from April 2007 to March 2015. We excluded patients with cerebral palsy, Down syndrome, or congenital disorders. In 2018, we retrospectively reviewed the clinical charts of the enrolled children. We investigated family history, neurological findings, and the developmental outcome during the follow-up period. RESULTS: During the follow-up period, 20 children (42%) were diagnosed with ASD. Gross motor development in infancy was not different between infants with and without ASD. The rate of poor eye contact at the first visit and a delay in the first word speech were significantly higher in infants with ASD than in infants without ASD. A family history of bottom shuffling was significantly less frequent in infants with ASD (10%) than in those without (39%). CONCLUSION: Some of bottom shufflers may represent ASD during follow-up. Paying attention to social and cognitive functions in shuffling babies is important.

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  243. 術前診断にて良性腫瘍と考えられたが全摘出できなかった腰髄腫瘍の2歳男児

    吉田 太郎, 村松 秀城, 今屋 雅之, 山森 彩子, 若松 学, 三輪田 俊介, 成田 幸太郎, 北澤 宏展, 谷口 理恵子, 市川 大輔, 濱田 太立, 西川 英里, 川島 希, 成田 敦, 西尾 信博, 小島 勢二, 高橋 義行, 田中 智史, 安藤 圭, 小林 和克, 今釜 史郎, 中黒 匡人, 平戸 純子

    日本小児血液・がん学会雑誌   Vol. 57 ( 5 ) page: 409 - 410   2021.2

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  244. 胎児発育不全に伴う脳発達異常の早期診断法確立に向けたバイオマーカーの探索

    小野田 淳人, 北瀬 悠磨, 辻 雅弘, 上田 一仁, 高橋 義行, 早川 昌弘, 佐藤 義朗

    日本小児科学会雑誌   Vol. 125 ( 2 ) page: 216 - 216   2021.2

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  245. 化学療法による治療介入を行った生後2ヵ月未満発症の神経芽腫4S

    今屋 雅之, 西川 英里, 吉田 太郎, 山森 彩子, 若松 学, 三輪田 俊介, 成田 幸太郎, 北澤 宏展, 谷口 理恵子, 市川 大輔, 濱田 太立, 川島 希, 成田 敦, 村松 秀城, 西尾 信博, 小島 勢二, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 57 ( 5 ) page: 411 - 411   2021.2

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  246. Long-term outcome in patients with Fanconi anemia who received hematopoietic stem cell transplantation: a retrospective nationwide analysis.

    Miharu Yabe, Tomohiro Morio, Ken Tabuchi, Daisuke Tomizawa, Daiichiro Hasegawa, Hiroyuki Ishida, Nao Yoshida, Takashi Koike, Yoshiyuki Takahashi, Katsuyoshi Koh, Yasuhiro Okamoto, Hideki Sano, Keisuke Kato, Yoshinobu Kanda, Hiroaki Goto, Junko Takita, Takako Miyamura, Maiko Noguchi, Koji Kato, Yoshiko Hashii, Yoshiko Astuta, Hiromasa Yabe

    International journal of hematology   Vol. 113 ( 1 ) page: 134 - 144   2021.1

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    We retrospectively analyzed nationwide records of 163 Fanconi anemia (FA) patients [aplastic anemia (AA), n = 118; myelodysplastic syndrome (MDS), n = 30; acute leukemia, n = 15] who underwent first allogeneic hematopoietic stem cell transplantation (HSCT) between 1987 and 2015 in Japan. An alternative donor was used in 119 (73%) patients, and 160 (98%) patients received a non-T-cell-depleted graft. With an 8.7-year median follow-up, 5-year overall survival (OS) was 81%. The 5-year OS was significantly higher in AA patients than in MDS and acute leukemia patients (89%, 71%, and 44%, respectively). In the MDS/leukemia group, factors associated with poor outcome in univariate analysis were older age at HSCT (≥ 18 years), conditioning regimen without anti-thymocyte or lymphocyte globulin, and grade II-IV acute graft-versus-host disease. After 1 year, of 137 survivors, 15 developed subsequent malignancies, of whom 12 were diagnosed with head and neck (HN)/esophageal cancer. An irradiation regimen and older age were associated with the risk of HN/esophageal cancer. Five of seven deaths were attributed to subsequent malignancies more than 5 years after HSCT. On the basis of the risk factors for HSCT in MDS/leukemia patients and subsequent malignancies, a more effective HSCT approach is required.

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  247. Detection of subclonal SETBP1 and JAK3 mutations in juvenile myelomonocytic leukemia using droplet digital PCR. Reviewed International journal

    Manabu Wakamatsu, Yusuke Okuno, Norihiro Murakami, Shunsuke Miwata, Hironobu Kitazawa, Kotaro Narita, Shinsuke Kataoka, Daisuke Ichikawa, Motoharu Hamada, Rieko Taniguchi, Kyogo Suzuki, Nozomu Kawashima, Eri Nishikawa, Atsushi Narita, Nobuhiro Nishio, Seiji Kojima, Hideki Muramatsu, Yoshiyuki Takahashi

    Leukemia   Vol. 35 ( 1 ) page: 259 - 263   2021.1

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    DOI: 10.1038/s41375-020-0817-x

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  248. A Breast-Fed Baby with Low KREC in TREC/KREC Newborn Screening Whose Mother Received Azathioprine Treatment

    Manabu Wakamatsu, Hideki Muramatsu, Daiei Kojima, Yusuke Okuno, Yoshimi Sakai, Yoko Nakajima, Tetsuya Ito, Nozomu Kawashima, Atsushi Narita, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  249. Allogeneic Stem Cell Transplantation from Alternative Donors for Children with Aplastic Anemia

    Atsushi Narita, Hideki Muramatsu, Masayuki Imaya, Ayako Yamamori, Taro Yoshida, Manabu Wakamatsu, Shunsuke Miwata, Koutaro Narita, Hironobu Kitazawa, Rieko Taniguchi, Motoharu Hamada, Daisuke Ichikawa, Eri Nishikawa, Nozomu Kawashima, Yusuke Okuno, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    BONE MARROW TRANSPLANTATION   Vol. 55 ( SUPPL 1 ) page: 215 - 216   2020.12

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  250. Allogeneic Hematopoietic Cell Transplantation in First Remission in Children with AML: A Comparison of RIC and MAC

    Asahito Hama, Daiki Yamashita, Daichi Sajiki, Hironobu Kitazawa, Hirotoshi Sakaguchi, Nao Yoshida, Nozomu Kawashima, Atsushi Narita, Hideki Muramatsu, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  251. Clinical Features of Cord Colitis Syndrome in Children

    Ryo Maemura, Atsushi Narita, Daisuke Ichikawa, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Nobuhiro Nishio, Hideki Muramatsu, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  252. Biomarkers for Distinguishing between acute GVHD and HHV-6B-Associated Skin Rash after Hematopoietic Stem Cell Transplantation

    Miura, H; Kudo, K; Tanaka, M; Takahashi, Y; Kojima, S; Yoshikawa, T

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  253. INFANTILE HEMISPHERIC BRAIN TUMOR WITH A GOPC-ROS1 FUSION GENE: A CASE REPORT

    Rieko Taniguchi, Atsushi Natsume, Nozomu Kawashima, Shinji Tanioka, Hideki Muramatsu, Motoharu Hamada, Daisuke Ichikawa, Masayuki Imaya, Kotaro Narita, Michihiro Kurimoto, Yoshiki Shiba, Kosuke Aoki, Fumiharu Ohka, Junko Hirato, Toshihiko Wakabayashi, Yoshiyuki Takahashi

    NEURO-ONCOLOGY   Vol. 22   page: 377 - 377   2020.12

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  254. Digenic mutations in ALDH2 and ADH5 impair formaldehyde clearance and cause a multisystem disorder, AMeD syndrome

    Yasuyoshi Oka, Motoharu Hamada, Yuka Nakazawa, Hideki Muramatsu, Yusuke Okuno, Koichiro Higasa, Mayuko Shimada, Honoka Takeshima, Katsuhiro Hanada, Taichi Hirano, Toshiro Kawakita, Hirotoshi Sakaguchi, Takuya Ichimura, Shuichi Ozono, Kotaro Yuge, Yoriko Watanabe, Yuko Kotani, Mutsumi Yamane, Yumiko Kasugai, Miyako Tanaka, Takayoshi Suganami, Shinichiro Nakada, Norisato Mitsutake, Yuichiro Hara, Kohji Kato, Seiji Mizuno, Noriko Miyake, Yosuke Kawai, Katsushi Tokunaga, Masao Nagasaki, Seiji Kito, Keiichi Isoyama, Masafumi Onodera, Hideo Kaneko, Naomichi Matsumoto, Fumihiko Matsuda, Keitaro Matsuo, Yoshiyuki Takahashi, Tomoji Mashimo, Seiji Kojima, Tomoo Ogi

    Science Advances   Vol. 6 ( 51 ) page: eabd7197 - eabd7197   2020.12

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    Rs671 in the aldehyde dehydrogenase 2 gene (<italic>ALDH2</italic>) is the cause of Asian alcohol flushing response after drinking. ALDH2 detoxifies endogenous aldehydes, which are the major source of DNA damage repaired by the Fanconi anemia pathway. Here, we show that the rs671 defective allele in combination with mutations in the alcohol dehydrogenase 5 gene, which encodes formaldehyde dehydrogenase (<italic>ADH5<sup>FDH</sup></italic>), causes a previously unidentified disorder, AMeD (aplastic anemia, mental retardation, and dwarfism) syndrome. Cellular studies revealed that a decrease in the formaldehyde tolerance underlies a loss of differentiation and proliferation capacity of hematopoietic stem cells. Moreover, <italic>Adh5<sup>−/−</sup>Aldh2</italic><sup>E506K/E506K</sup> double-deficient mice recapitulated key clinical features of AMeDS, showing short life span, dwarfism, and hematopoietic failure. Collectively, our results suggest that the combined deficiency of formaldehyde clearance mechanisms leads to the complex clinical features due to overload of formaldehyde-induced DNA damage, thereby saturation of DNA repair processes.

    DOI: 10.1126/sciadv.abd7197

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  255. Development of Donor-Derived T cell EBV-LPD with Identical EBV Genome after Allogeneic CBT in the NK Cell-Type CAEBV Patient

    Masayuki Imaya, Yusuke Okuno, Nozomu Kawashima, Manabu Wakamatsu, Hideki Muramatsu, Nobuhiro Nishio, Akihisa Sawada, Junichi Kawada, Yoshinori Ito, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  256. Effect of Azacitidine in a Child with Fanconi Anemia and Myelodysplastic Syndrome Harboring Monosomy 7

    Nozomu Kawashima, Masafumi Ito, Kotaro Narita, Daisuke Ichikawa, Eri Nishikawa, Atsushi Narita, Hideki Muramatsu, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  257. Hematopoietic Stem Cell Transplantation for Severe Combined Immunodeficiency in Japan: A Nationwide Retrospective Analysis

    Miyamoto, S; Yanagimachi, M; Umeda, K; Iguchi, A; Sasahara, Y; Takada, H; Takahashi, Y; Kajiwara, M; Inoue, M; Kato, K; Hashii, Y; Atsuta, Y; Yabe, H; Imai, K; Morio, T

    BONE MARROW TRANSPLANTATION   Vol. 55 ( SUPPL 1 ) page: 443 - 443   2020.12

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  258. Hematopoietic Stem Cell Transplantation for Inborn Errors of Immunity in Japan: Overview of a Nationwide Retrospective Analysis

    Miyamoto, S; Yanagimachi, M; Umeda, K; Iguchi, A; Sasahara, Y; Takada, H; Yamada, M; Ishimura, M; Koike, T; Yasui, M; Takahashi, Y; Kawaguchi, H; Kajiwara, M; Inoue, M; Kato, K; Hashii, Y; Atsuta, Y; Yabe, H; Imai, K; Morio, T

    BONE MARROW TRANSPLANTATION   Vol. 55 ( SUPPL 1 ) page: 102 - 103   2020.12

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  259. Favorable Prognosis of Patients with Intermediate-Risk Neuroblastoma Having Residual Tumor after Treatment

    Nobuhiro Nishio, Daisuke Ichikawa, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Atsushi Narita, Hideki Muramatsu, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  260. Viral Reactivations in Children with Aplastic Anemia Undergoing Allogeneic Hematopoietic Stem Cell Transplantation

    Atsushi Narita, Yoshinori Ito, Nozomu Kawashima, Yusuke Okuno, Hideki Muramatsu, Nobuhiro Nishio, Yuka Torii, Junichi Kawada, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  261. Microsatellite Instability-High is Rare Events in Refractory Pediatric Solid Tumors

    Yoshida, T; Muramatsu, H; Wakamatsu, M; Taniguchi, R; Ichikawa, D; Nakaguro, M; Natsume, A; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  262. Reduced Intensity Conditioning is Effective for Hematopoietic stem Cell Transplantation in Patients with Diamond-Blackfan ANEMIA

    Koyamaishi, S; Kamio, T; Kobayashi, A; Sato, T; Kudo, K; Sasaki, S; Kanezaki, R; Hasegawa, D; Muramatsu, H; Takahashi, Y; Sasahara, Y; Hiramatsu, H; Kakuda, H; Tanaka, M; Ishimura, M; Nishi, M; Ishiguro, A; Yabe, H; Yoshida, K; Kanno, H; Ohga, S; Ohara, A; Kojima, S; Miyano, S; Ogawa, S; Toki, T; Terui, K; Ito, E

    BONE MARROW TRANSPLANTATION   Vol. 55 ( SUPPL 1 ) page: 36 - 37   2020.12

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  263. Strategy of Stem Cell Transplantation at 1st Molecular CR for Pediatric Acute Lymphoblastic Leukemia with MEF2D-BCL9 Fusion

    Motoharu Hamada, Masayuki Imaya, Yusuke Okuno, Daisuke Ichikawa, Nozomu Kawashima, Atsushi Narita, Eri Nishikawa, Hideki Muramatsu, Nobuhiro Nishio, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 67   2020.12

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  264. Myelodysplastic syndromes in a pediatric patient with Cri du Chat syndrome with a ring chromosome 5. Reviewed

    Akifumi Nozawa, Michio Ozeki, Shiho Yasue, Saori Endo, Tomonori Kadowaki, Hidenori Ohnishi, Hideki Muramatsu, Asahito Hama, Yoshiyuki Takahashi, Seiji Kojima, Toshiyuki Fukao

    International journal of hematology   Vol. 112 ( 5 ) page: 728 - 733   2020.11

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    Few hematological complications have previously been reported in association with Cri du Chat syndrome (CdCS). A case of myelodysplastic syndromes (MDS) in a pediatric patient with CdCS is herein presented. A 17-year-old female with CdCS caused by ring chromosome 5 was admitted to the hospital for investigation of a 1-month history of anemia. Based on the morphological findings of bone marrow, the patient was diagnosed with refractory cytopenia with multilineage dysplasia. The risk group was classified as intermediate-1 in the International Prognostic Scoring System (IPSS), and low in the revised IPSS. Assessment by microarray comparative genomic hybridization (CGH) identified the breakpoints of ring chromosome 5 as 46,XX,r(5)(p14.3q35.3). This revealed that the 5q terminal deletion did not include the common deleted region of MDS with del(5q). Treatment with azacitidine was initiated to control disease progression and improve quality of life. At baseline, the patient had a mean transfusion requirement of 3 units/month, which decreased to 2 units/month after six cycles of azacitidine and to 1 unit/month after 10 cycles of azacitidine. Cytopenia observed in the presented case seemed irrelevant to ring chromosome 5 which is the causative cytogenetic abnormality of CdCS, and further analyses may be needed to clarify the pathogenesis.

    DOI: 10.1007/s12185-020-02909-7

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  265. Risk factors for secondary poor graft function after bone marrow transplantation in children with acquired aplastic anemia Reviewed International journal

    Asahito Hama, Hideki Muramatsu, Atsushi Narita, Eri Nishikawa, Nozomu Kawashima, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    Pediatric transplantation   Vol. 24 ( 7 ) page: e13828   2020.11

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    In patients with acquired AA, PGF is a major cause of cytopenia after hematopoietic stem cell transplantation. An increased incidence of PGF, especially sPGF, has been noted after the introduction of the FLU/CY regimen in children with acquired AA. To clarify the risk factors for sPGF, the clinical data of 49 patients (median age, 11 years; range, 1-19 years) with AA who received allogeneic BMT at Nagoya University Hospital from 1997 to 2016 were analyzed. Out of the 49 patients, 7 developed sPGF, and the 5-year CI was 0.15 (95% CI, 0.04-0.25). Five received the FLU/CY regimen, and the 5-year CI of sPGF was significantly higher in patients who received the regimen (0.36; 95% CI, 0.12-0.62) than in those who were conditioned with the non-FLU/CY regimen (0.06; 95% CI, 0.01-0.17; P =.01). The multivariate analysis confirmed that the FLU/CY regimen (hazard ratio, 6.12; 95% CI, 1.16-32.4; P =.03) was a significant risk factor for sPGF. sPGF improved spontaneously without stem cell boost infusions in 5 patients, ranging from 460 to 3539 days after BMT. The 10-year CI of the spontaneous trilineage recovery was 0.83 (95% CI, 0.00-0.97), and all 7 patients are alive. The FLU/CY regimen was identified as a risk factor for the sPGF development in patients with AA. The establishment of the optimal conditioning regimens for children with AA is warranted.

    DOI: 10.1111/petr.13828

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  266. 造血細胞移植後小児のキャッチアップ接種と移植細胞ソース別の比較検討

    鈴木 高子, 川田 潤一, 西川 英里, 山口 慎, 奥村 俊彦, 武内 俊, 堀場 千尋, 鳥居 ゆか, 村松 秀城, 高橋 義行, 伊藤 嘉規

    日本小児感染症学会総会・学術集会プログラム・抄録集   Vol. 52回   page: 179 - 179   2020.11

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  267. 分子医学・再生医療・心臓血管発生「先天性心疾患の理解・治療・予防につなげる臨床心臓発生学」 ABL1変異は先天性心疾患を伴う症候群の原因となる エクソーム解析と質量分析法を応用した遺伝性心室中隔欠損症の網羅的病態解明

    山本 英範, 早野 聡, 奥野 友介, 小野田 淳人, 加藤 耕治, 長井 典子, 深澤 佳絵, 齋藤 伸治, 高橋 義行, 加藤 太一

    日本小児循環器学会雑誌   Vol. 36 ( Suppl.2 ) page: s2 - 118   2020.11

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  268. Comprehensive pathogen detection in sera of Kawasaki disease patients by high-throughput sequencing: a retrospective exploratory study. International journal

    Yuka Torii, Kazuhiro Horiba, Satoshi Hayano, Taichi Kato, Takako Suzuki, Jun-Ichi Kawada, Yoshiyuki Takahashi, Seiji Kojima, Yusuke Okuno, Tomoo Ogi, Yoshinori Ito

    BMC pediatrics   Vol. 20 ( 1 ) page: 482 - 482   2020.10

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    BACKGROUND: Kawasaki disease (KD) is an idiopathic systemic vasculitis that predominantly damages coronary arteries in children. Various pathogens have been investigated as triggers for KD, but no definitive causative pathogen has been determined. As KD is diagnosed by symptoms, several days are needed for diagnosis. Therefore, at the time of diagnosis of KD, the pathogen of the trigger may already be diminished. The aim of this study was to explore comprehensive pathogens in the sera at the acute stage of KD using high-throughput sequencing (HTS). METHODS: Sera of 12 patients at an extremely early stage of KD and 12 controls were investigated. DNA and RNA sequences were read separately using HTS. Sequence data were imported into the home-brew meta-genomic analysis pipeline, PATHDET, to identify the pathogen sequences. RESULTS: No RNA virus reads were detected in any KD case except for that of equine infectious anemia, which is known as a contaminant of commercial reverse transcriptase. Concerning DNA viruses, human herpesvirus 6B (HHV-6B, two cases) and Anelloviridae (eight cases) were detected among KD cases as well as controls. Multiple bacterial reads were obtained from KD and controls. Bacteria of the genera Acinetobacter, Pseudomonas, Delfita, Roseomonas, and Rhodocyclaceae appeared to be more common in KD sera than in the controls. CONCLUSION: No single pathogen was identified in serum samples of patients at the acute phase of KD. With multiple bacteria detected in the serum samples, it is difficult to exclude the possibility of contamination; however, it is possible that these bacteria might stimulate the immune system and induce KD.

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  269. MicroRNA-155-5p Plays a Critical Role in Transient Leukemia of Down Syndrome by Targeting Tumor Necrosis Factor Receptor Superfamily Members.

    Sas V, Pasca S, Jurj A, Pop L, Muramatsu H, Ono H, Dima D, Teodorescu P, Iluta S, Turcas C, Onaciu A, Munteanu R, Zimta AA, Blag C, Popa G, von Gamm EDA, Arghirescu S, Serban M, Man S, Marian M, Petrushev B, Berce C, Colita A, Zdrenghea M, Kojima S, Gulei D, Takahashi Y, Tomuleasa C

    Cellular physiology and biochemistry : international journal of experimental cellular physiology, biochemistry, and pharmacology   Vol. 54 ( 5 ) page: 994 - 1012   2020.10

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    DOI: 10.33594/000000283

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  270. Change of White Matter Integrity in Children With Hematopoietic Stem Cell Transplantation. International journal

    Yoko Sakaguchi, Jun Natsume, Hiroyuki Kidokoro, Masaharu Tanaka, Yu Okai, Yuji Ito, Hiroyuki Yamamoto, Atsuko Ohno, Tomohiko Nakata, Toshiki Nakane, Hisashi Kawai, Toshiaki Taoka, Hideki Muramatsu, Shinji Naganawa, Yoshiyuki Takahashi

    Pediatric neurology   Vol. 111   page: 78 - 84   2020.10

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    BACKGROUND: Advances in hematopoietic stem cell transplantation have improved the survival rate of malignant diseases and congenital immunodeficiencies. It has become important to assess long-term complications in survivors. To assess neurological abnormalities in children treated by transplantation, diffusion tensor imaging was performed. METHODS: Forty children who underwent head diffusion tensor imaging before and after their first transplantation were enrolled. Patients with brain lesions on conventional MRI were excluded. Fractional anisotropy and mean diffusivity were compared between patients and 28 control subjects using tract-based spatial statistics. The Strengths and Difficulties Questionnaire was administered as a behavioral evaluation after transplantation, and diffusion tensor images of patients with and without behavioral abnormalities were compared. RESULTS: The age of patients and controls was 0 to 19 years and 0 to 16 years, respectively. The date of diffusion tensor imaging was 10 to 57 days before and 40 to 153 days after transplantation. Tract-based spatial statistics showed fractional anisotropy reduction in widespread white matter in patients before and after transplantation. Mean diffusivity was high before transplantation and normalized after transplantation. Analysis comparing before and after hematopoietic stem cell transplantation shows no difference in fractional anisotropy and a higher mean diffusivity before hematopoietic stem cell transplantation. In patients with behavioral abnormalities, low fractional anisotropy and high mean diffusivity remained after transplantation. CONCLUSIONS: Longitudinal diffusion tensor imaging showed white matter abnormalities in children without conventional MRI abnormalities, which were related to behavioral problems after transplantation. Diffusion tensor imaging is useful for behavioral assessment in children undergoing transplantation.

    DOI: 10.1016/j.pediatrneurol.2020.06.008

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  271. Successful treatment with i.v. immunoglobulin and rituximab for bronchiolitis obliterans associated with paraneoplastic pemphigus. Reviewed International journal

    Kaori Kaibuchi-Ando, Kazumitsu Sugiura, Yoshinao Muro, Yoshiyuki Takahashi, Seiji Kojima, Norito Ishii, Takashi Hashimoto, Masashi Akiyama

    The Journal of dermatology   Vol. 47 ( 10 ) page: E368 - E370   2020.10

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    DOI: 10.1111/1346-8138.15496

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  272. 臍帯血移植後、ドナー細胞に移植前と同一のEBV感染が確認された慢性活動性EBV感染症の再発例(Development of donor-derived T cell EBV-LPD with identical EBV genome after allogeneic CBT in the NK cell-type CAEBV patient)

    Imaya Masayuki, Okuno Yusuke, Kawashima Nozomu, Wakamatsu Manabu, Muramatsu Hideki, Nishio Nobuhiro, Sawada Akihisa, Kawada Junichi, Ito Yoshinori, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 261 - 261   2020.10

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  273. 治療後に腫瘍が残存している中間リスク神経芽腫患者の良好な予後(Favorable prognosis of patients with intermediate-risk neuroblastoma having residual tumor after treatment)

    Nishio Nobuhiro, Ichikawa Daisuke, Hamada Motoharu, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 251 - 251   2020.10

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  274. 小児急性骨髄性白血病第一寛解期における同種造血細胞移植 RICとMACの比較(Allogeneic hematopoietic cell transplantation in first remission in children with AML: A comparison of RIC and MAC)

    Hama Asahito, Yamashita Daiki, Sajiki Daichi, Kitazawa Hironobu, Sakaguchi Hirotoshi, Yoshida Nao, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 221 - 221   2020.10

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  275. 小児におけるcord colitis syndromeの臨床的特徴(Clinical features of cord colitis syndrome in children)

    Maemura Ryo, Narita Atsushi, Ichikawa Daisuke, Hamada Motoharu, Nishikawa Eri, Kawashima Nozomu, Nishio Nobuhiro, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 261 - 261   2020.10

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  276. 小児再生不良性貧血における同種造血細胞移植後のウイルス再活性化(Viral reactivations in children with aplastic anemia undergoing allogeneic hematopoietic stem cell transplantation)

    Narita Atsushi, Ito Yoshinori, Kawashima Nozomu, Okuno Yusuke, Muramatsu Hideki, Nishio Nobuhiro, Torii Yuka, Kawada Junichi, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 235 - 235   2020.10

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  277. ファンコニ貧血を背景にモノソミー7を有する骨髄異形成症候群に対するアザシチジンの効果(Effect of azacitidine in a child with Fanconi anemia and myelodysplastic syndrome harboring monosomy 7)

    Kawashima Nozomu, Ito Masafumi, Narita Kotaro, Ichikawa Daisuke, Nishikawa Eri, Narita Atsushi, Muramatsu Hideki, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 233 - 233   2020.10

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  278. アザチオプリン内服中の母体から出生し、新生児マススクリーニングでKREC低値を認めた一例(A breast-fed baby with low KREC in TREC/KREC newborn screening whose mother received azathioprine treatment)

    Wakamatsu Manabu, Muramatsu Hideki, Kojima Daiei, Okuno Yusuke, Sakai Yoshimi, Nakajima Yoko, Ito Tetsuya, Kawashima Nozomu, Narita Atsushi, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 241 - 241   2020.10

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  279. MEF2D-BCL9融合遺伝子陽性小児ALLに対する第一寛解期造血幹細胞移植(Strategy of stem cell transplantation at 1st molecular CR for pediatric acute lymphoblastic leukemia with MEF2D-BCL9 fusion)

    Hamada Motoharu, Imaya Masayuki, Okuno Yusuke, Ichikawa Daisuke, Kawashima Nozomu, Narita Atsushi, Nishikawa Eri, Muramatsu Hideki, Nishio Nobuhiro, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 57 ( 4 ) page: 230 - 230   2020.10

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  280. Direct Delivery of piggyBac CD19 CAR T Cells Has Potent Anti-tumor Activity against ALL Cells in CNS in a Xenograft Mouse Model. Reviewed International journal

    Kuniaki Tanaka, Itaru Kato, Miyuki Tanaka, Daisuke Morita, Kazuyuki Matsuda, Yoshiyuki Takahashi, Tatsutoshi Nakahata, Katsutsugu Umeda, Hidefumi Hiramatsu, Souichi Adachi, Junko Takita, Yozo Nakazawa

    Molecular therapy oncolytics   Vol. 18   page: 37 - 46   2020.9

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    The anti-CD19 chimeric antigen receptor (CAR) T cells showed excellent effect against acute lymphoblastic leukemia (ALL) in bone marrow (BM) in clinical trials. However, it remains to be elucidated whether the CD19 CAR T cell therapy is effective for ALL cells in central nervous system (CNS) because the patients with isolated or advanced CNS disease were excluded from clinical trials of systemic intravenous (i.v.) delivery of CAR T cells. Therefore, the preclinical evaluation for the efficacy of CAR T cell therapy against ALL cells in CNS is essential for clinical application. We evaluated the effect and adverse reaction of CD19 CAR T cells against ALL in CNS using a xenograft mouse model by i.v. or intra-cerebroventricular (i.c.v.) delivery of CAR T cells. Injection of piggyBac CD19 CAR T cells by i.v. had partial effects, whereas all CAR T i.c.v.-delivered mice had eliminated ALL in CNS. Although some CAR T i.c.v.-delivered mice showed transient changes of clinical symptoms during the first few days after treatment, none of CAR T i.c.v.-delivered mice displayed fatal adverse events. In this study, we demonstrated that direct delivery into CNS of CAR T cells is a possible therapeutic approach with the xenograft mouse model.

    DOI: 10.1016/j.omto.2020.05.013

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  281. Association between graft source and response to live-attenuated vaccination in pediatric hematopoietic stem cell transplantation recipients: a single-center retrospective study. Reviewed International journal

    Takako Suzuki, Jun-Ichi Kawada, Eri Nishikawa, Yuka Torii, Kazuhiro Horiba, Suguru Takeuchi, Toshihiko Okumura, Hideki Muramatsu, Yoshiyuki Takahashi, Yoshinori Ito

    Bone marrow transplantation   Vol. 55 ( 9 ) page: 1872 - 1874   2020.9

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    DOI: 10.1038/s41409-020-0867-8

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  282. Establishment of a Novel Fetal Growth Restriction Model and Development of a Stem-Cell Therapy Using Umbilical Cord-Derived Mesenchymal Stromal Cells. International journal

    Yuma Kitase, Yoshiaki Sato, Sakiko Arai, Atsuto Onoda, Kazuto Ueda, Shoji Go, Haruka Mimatsu, Mahboba Jabary, Toshihiko Suzuki, Miharu Ito, Akiko Saito, Akihiro Hirakawa, Takeo Mukai, Tokiko Nagamura-Inoue, Yoshiyuki Takahashi, Masahiro Tsuji, Masahiro Hayakawa

    Frontiers in cellular neuroscience   Vol. 14   page: 212 - 212   2020.7

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    Fetal growth restriction (FGR) is a major complication of prenatal ischemic/hypoxic exposure and affects 5%-10% of pregnancies. It causes various disorders, including neurodevelopmental disabilities due to chronic hypoxia, circulatory failure, and malnutrition via the placenta, and there is no established treatment. Therefore, the development of treatments is an urgent task. We aimed to develop a new FGR rat model with a gradual restrictive load of uterus/placental blood flow and to evaluate the treatment effect of the administration of umbilical cord-derived mesenchymal stromal cells (UC-MSCs). To create the FGR rat model, we used ameroid constrictors that had titanium on the outer wall and were composed of C-shaped casein with a notch and center hole inside that gradually narrowed upon absorbing water. The ameroid constrictors were attached to bilateral ovarian/uterine arteries on the 17th day of pregnancy to induce chronic mild ischemia, which led to FGR with over 20% bodyweight reduction. After the intravenous administration of 1 × 105 UC-MSCs, we confirmed a significant improvement in the UC-MSC group in a negative geotaxis test at 1 week after birth and a rotarod treadmill test at 5 months old. In the immunobiological evaluation, the total number of neurons counted via the stereological counting method was significantly higher in the UC-MSC group than in the vehicle-treated group. These results indicate that the UC-MSCs exerted a treatment effect for neurological impairment in the FGR rats.

    DOI: 10.3389/fncel.2020.00212

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  283. Lower limb pain following allogeneic hematopoietic stem cell transplantation in Japanese children. Reviewed

    Kenichi Mishima, Hiroshi Kitoh, Masaki Matsushita, Tadashi Nagata, Yasunari Kamiya, Yoshiyuki Takahashi, Naoki Ishiguro

    Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association   Vol. 25 ( 4 ) page: 682 - 687   2020.7

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    BACKGROUND: Patients who undergo allogeneic hematopoietic stem cell transplantation (allo-HSCT) can experience musculoskeletal pains in their lower limbs in the early and late post-transplantation period. This study investigated demographics and clinical characteristics of the lower limb pain (LLP) among Japanese survivors of pediatric allo-HSCT. METHODS: A total of 143 consecutive Japanese patients who had undergone allo-HSCT less than 18 years of age in a single institute between 2005 and 2015 were reviewed. Patients referred for in-house orthopedic evaluation of their sustained LLPs that impaired ambulation were defined as LLP group. Illness/transplantation-related parameters were compared between LLP group and non-LLP group. RESULTS: Ninety children with a mean age of 8.5 years at transplantation were enrolled. Their median follow-up period following transplantation was 6.3 years (range, 2.1-12.5). There were four patients in LLP group, whose etiologies were AVN of the femoral head and insufficiency fracture (ISF) of the tibia or the medial cuneiform bone. Cumulative dose of steroids that administered from six months before transplantation to six months after discharge from hospitalization for transplantation was significantly higher in LLP group than non-LLP group. Additionally, the two groups differed significantly in terms of hospitalization period after transplantation. LLP caused by AVN of the femoral head manifested between six months and two years, whereas that caused by ISF within the first six months after transplantation. CONCLUSIONS: The incidence of sustained LLP that impairs ambulation following contemporary allo-HSCT is not common in Japanese pediatric survivors. The risk of developing musculoskeletal LLP may increase with a higher steroid dosage in the peri-transplant period. LLP caused by AVN of the femoral head is likely to manifest later than that caused by ISF.

    DOI: 10.1016/j.jos.2019.07.007

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  284. Conditioning regimen for allogeneic bone marrow transplantation in children with acquired bone marrow failure: fludarabine/melphalan vs. fludarabine/cyclophosphamide. Reviewed International journal

    Nao Yoshida, Yoshiyuki Takahashi, Hiromasa Yabe, Ryoji Kobayashi, Kenichiro Watanabe, Kazuko Kudo, Miharu Yabe, Takako Miyamura, Katsuyoshi Koh, Hiroshi Kawaguchi, Hiroaki Goto, Naoto Fujita, Keiko Okada, Yasuhiro Okamoto, Koji Kato, Masami Inoue, Ritsuro Suzuki, Yoshiko Atsuta, Seiji Kojima

    Bone marrow transplantation   Vol. 55 ( 7 ) page: 1272 - 1281   2020.7

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    Fludarabine/cyclophosphamide-based conditioning regimens are standard in bone marrow transplantation (BMT) for acquired bone marrow failure in children, however, graft failure may occur. Using the data from a nationwide transplantation registry, we compared the outcomes of children aged <16 years with acquired aplastic anemia and refractory cytopenia of childhood who underwent allogeneic BMT with either fludarabine/melphalan (n = 71) or fludarabine/cyclophosphamide (n = 296) between 2000 and 2016. The fludarabine/melphalan regimen provided excellent outcomes, with 3-year overall survival and failure-free survival rates of 98% and 97%, respectively. The 83% 3-year failure-free survival in the fludarabine/cyclophosphamide group was significantly inferior (P = 0.002), whereas the overall survival did not differ between the two groups. Late graft failure was the most common cause of treatment failure in the fludarabine/cyclophosphamide group, which experienced a significantly higher incidence of late graft failure than the fludarabine/melphalan group (11% vs. 3%; P = 0.035). Multivariate analyses showed that the fludarabine/melphalan regimen was associated with a better failure-free survival (hazard ratio [HR] 0.12; P = 0.005) and lower risk of late graft failure (HR 0.16; P = 0.037). Fludarabine/melphalan-based conditioning regimen can be a promising option for children with acquired bone marrow failure receiving BMT.

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  285. Targeting MEF2D-fusion Oncogenic Transcriptional Circuitries in B-cell Precursor Acute Lymphoblastic Leukemia

    Tsuzuki, S; Yasuda, T; Kojima, S; Kawazu, M; Akahane, K; Inukai, T; Imaizumi, M; Morishita, T; Miyamura, K; Ueno, T; Karnan, S; Ota, A; Hyodo, T; Konishi, H; Sanada, M; Nagai, H; Horibe, K; Tomita, A; Suzuki, K; Muramatsu, H; Takahashi, Y; Miyazaki, Y; Matsumura, I; Kiyoi, H; Hosokawa, Y; Mano, H; Hayakawa, F

    BLOOD CANCER DISCOVERY   Vol. 1 ( 1 ) page: 82 - 95   2020.7

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    DOI: 10.1158/2643-3249.BCD-19-0080

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  286. Targeting MEF2D-fusion Oncogenic Transcriptional Circuitries in B-cell Precursor Acute Lymphoblastic Leukemia Reviewed

    Shinobu Tsuzuki, Takahiko Yasuda, Shinya Kojima, Masahito Kawazu, Koshi Akahane, Takeshi Inukai, Masue Imaizumi, Takanobu Morishita, Koichi Miyamura, Toshihide Ueno, Sivasundaram Karnan, Akinobu Ota, Toshinori Hyodo, Hiroyuki Konishi, Masashi Sanada, Hirokazu Nagai, Keizo Horibe, Akihiro Tomita, Kyogo Suzuki, Hideki Muramatsu, Yoshiyuki Takahashi, Yasushi Miyazaki, Itaru Matsumura, Hitoshi Kiyoi, Yoshitaka Hosokawa, Hiroyuki Mano, Fumihiko Hayakawa

    Blood Cancer Discovery   Vol. 1 ( 1 ) page: 82 - 95   2020.7

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    DOI: 10.1158/2643-3230.BCD-19-0080

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  287. 【骨髄不全症の病態と検査】骨髄不全症と遺伝子異常 血球テロメア長測定の臨床的意義

    成田 敦, 高橋 義行

    臨床検査   Vol. 64 ( 7 ) page: 761 - 765   2020.7

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    <文献概要>Point ●再生不良性貧血(AA)を含む骨髄不全症患者ではテロメア長の短縮がみられる.造血ストレスに伴う二次性の変化が原因と考えられている.●骨髄不全症患者の一部にはテロメラーゼ複合体遺伝子群の変異を有する患者がいる.診断補助としてテロメア長の測定が有用である.●特徴的な身体症状がみられない不全型の先天性角化不全症(DC)においてはテロメア長の測定や遺伝子診断が必須である.

    Other Link: https://search-tp.jamas.or.jp/index.php?module=Default&action=Link&pub_year=2020&ichushi_jid=J01541&link_issn=&doc_id=20200714070008&doc_link_id=10.11477%2Fmf.1542202408&url=https%3A%2F%2Fdoi.org%2F10.11477%2Fmf.1542202408&type=%88%E3%8F%91.jp_%83I%81%5B%83%8B%83A%83N%83Z%83X&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00024_2.gif

  288. Safe diagnostic management of malignant mediastinal tumors in the presence of respiratory distress: a 10-year experience. Reviewed International journal

    Tomoko Tanaka, Hizuru Amano, Yujiro Tanaka, Yoshiyuki Takahashi, Tatsuro Tajiri, Takahisa Tainaka, Chiyoe Shirota, Wataru Sumida, Kazuki Yokota, Satoshi Makita, Yukiko Tani, Akinari Hinoki, Hiroo Uchida

    BMC pediatrics   Vol. 20 ( 1 ) page: 292 - 292   2020.6

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    BACKGROUND: The fundamental treatment for patients with pediatric malignant mediastinal tumors is chemotherapy. Therefore, accurate diagnosis is essential for selecting the appropriate chemotherapeutic regimen. However, malignant mediastinal tumors occasionally cause respiratory distress, and biopsies under general anesthesia are dangerous for such patients as invasive mechanical ventilation can aggravate airway obstruction caused by mass effect. In this study, we reviewed our 10-year diagnostic experience to evaluate the efficacy of our practices and confirm a safe diagnostic protocol for future patients. METHODS: We retrospectively reviewed medical records of children with malignant mediastinal tumors diagnosed at Nagoya University Hospital from 2007 to 2018 who demonstrated respiratory distress. Respiratory distress included dyspnea, massive pleural effusion, wheezing, and hypoxemia owing to tumors. Data on sex, age at onset, primary symptoms, location of tumor, management strategy (especially the method of diagnosis and definitive diagnosis), clinical course, prognosis during the acute phase (within 3 months from the onset of respiratory symptoms), and long-term outcome were collected. RESULTS: Twelve pediatric patients met the review criteria. There were seven anterior mediastinal tumors and five posterior mediastinal tumors. All anterior mediastinal tumors were diagnosed via bone marrow smear, thoracentesis, or core needle biopsy while maintaining spontaneous breathing. Regarding posterior tumors, two patients were diagnosed via a core needle biopsy and lymph node excisional biopsy under spontaneous breathing. Two cases were initially diagnosed solely using tumor markers. One patient with severe tracheal compression underwent tumor resection with extracorporeal membrane oxygenation stand-by. No patient died of diagnostic procedure-related complications. CONCLUSIONS: In 11 of the 12 cases reviewed, safe and accurate tumor diagnosis was accomplished without general anesthesia. A diagnostic strategy without general anesthesia considering the tumor location proved to be useful.

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  289. Impact of graft-versus-host disease on the clinical outcome of allogeneic hematopoietic stem cell transplantation for non-malignant diseases. Reviewed

    Katsutsugu Umeda, Kohsuke Imai, Masakatsu Yanagimachi, Hiromasa Yabe, Masao Kobayashi, Yoshiyuki Takahashi, Michiko Kajiwara, Nao Yoshida, Yuko Cho, Masami Inoue, Yoshiko Hashii, Yoshiko Atsuta, Tomohiro Morio

    International journal of hematology   Vol. 111 ( 6 ) page: 869 - 876   2020.6

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    The impact of acute and chronic graft-versus-host disease (GVHD) on clinical outcomes was retrospectively analyzed in 960 patients with non-malignant diseases (NMD) who underwent a first allogeneic hematopoietic stem cell transplantation (HSCT). Grade III-IV acute GVHD (but not grade I-II) was significantly associated with a lower rate of overall survival (OS), and higher non-relapse mortality (NRM) than that seen in patients without acute GVHD. Extensive (but not limited) GVHD was significantly associated with a lower OS rate and higher NRM than that seen in patients without chronic GVHD. Any grade of acute (but not chronic) GVHD was significantly associated with a lower incidence of relapse and a lower proportion of patients requiring a second HSCT or donor lymphocyte infusion for graft failure or mixed chimerism, but its impact on OS was almost negligible. Acute GVHD was significantly associated with lower OS rates in all disease groups, whereas chronic GVHD was significantly associated with lower OS rates in the primary immunodeficiency and histiocytosis groups. In conclusion, acute and chronic GVHD, even if mild, was associated with reduced OS in patients receiving HSCT for NMD and effective strategies should, therefore, be implemented to minimize GVHD.

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  290. A familial case of alveolar capillary dysplasia with misalignment of the pulmonary veins: the clinicopathological features and unusual glomeruloid endothelial proliferation. Reviewed International journal

    Akiko Kitano, Masato Nakaguro, Seiichi Tomotaki, Shintaro Hanaoka, Masahiko Kawai, Akiko Saito, Masahiro Hayakawa, Yoshiyuki Takahashi, Hidenori Kawasaki, Takahiro Yamada, Masahiko Ikeda, Tetsuo Onda, Kazutoshi Cho, Hironori Haga, Atsuko Nakazawa, Sachiko Minamiguchi

    Diagnostic pathology   Vol. 15 ( 1 ) page: 48 - 48   2020.5

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    BACKGROUND: Alveolar capillary dysplasia with misalignment of pulmonary veins (ACD/MPV) is a rare disorder of pulmonary vascular abnormality with persistent pulmonary hypertension of the newborn. The symptom usually presents within hours after birth, leading to an early demise. Heterozygous de novo point mutations and genomic deletions of the FOXF1 (forkhead box F1) gene or its upstream enhancer have been identified in most patients with ACD/MPV. Most cases of ACD/MPV are sporadic; however, familial cases are also reported in 10% of patients. CASE PRESENTATION: We herein report a case of familial ACD/MPV that showed unusual glomeruloid proliferation of endothelial cells. In this family, three of the four siblings died within two to 3 days after birth because of persistent pulmonary hypertension and respiratory failure. Only the second child remains alive and healthy. An autopsy was performed for the third and fourth children, resulting in a diagnosis of ACD/MPV based on the characteristic features, including misalignment of smaller pulmonary veins and lymphangiectasis. In both of these children, glomeruloid endothelial proliferation of vessels was noted in the interlobular septa. The vessels were immunohistochemically positive for D2-40, CD31, Factor VIII, and ERG, suggestive of differentiation for both lymphatic and blood vessels. CONCLUSIONS: Unusual glomeruloid endothelial proliferation was observed in a familial ACD/MPV case. This histologic feature has not been described previously in ACD/MPV or any other pulmonary disease. Although the histogenesis of this histologic feature is unclear, this finding may suggest that ACD/MPV is a compound vascular and lymphovascular system disorder that exhibits various histologic features.

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  291. Clinical Outcomes after Allogeneic Hematopoietic Stem Cell Transplantation in Children with Juvenile Myelomonocytic Leukemia: A Report from the Japan Society for Hematopoietic Cell Transplantation. Reviewed International journal

    Nao Yoshida, Hirotoshi Sakaguchi, Miharu Yabe, Daiichiro Hasegawa, Asahito Hama, Daisuke Hasegawa, Motohiro Kato, Maiko Noguchi, Kiminori Terui, Yoshiyuki Takahashi, Yuko Cho, Maho Sato, Katsuyoshi Koh, Harumi Kakuda, Hiroyuki Shimada, Yoshiko Hashii, Atsushi Sato, Koji Kato, Yoshiko Atsuta, Kenichiro Watanabe

    Biology of blood and marrow transplantation : journal of the American Society for Blood and Marrow Transplantation   Vol. 26 ( 5 ) page: 902 - 910   2020.5

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    Hematopoietic stem cell transplantation (HSCT) is the only curative treatment for juvenile myelomonocytic leukemia (JMML), but few large studies of HSCT for JMML exist. Using data from the Japan Society for Hematopoietic Cell Transplantation registry, we analyzed the outcomes of 129 children with JMML who underwent HSCT between 2000 and 2011. The 5-year overall survival (OS) rate and cumulative incidence of relapse were 64% and 34%, respectively. A regimen of busulfan/fludarabine/melphalan was the most commonly used (59 patients) and provided the best outcomes; the 5-year OS rate reached 73%, and the cumulative incidences of relapse and transplantation-related mortality were 26% and 9%, respectively. In contrast, the use of the irradiation-based myeloablative regimen was the most significant risk factor for OS (hazard ratio [HR], 2.92; P = .004) in the multivariate model. In addition, chronic graft-versus-host disease (GVHD) was strongly associated with lower relapse (HR, 0.37; P = .029) and favorable survival (HR, 0.22; P = .006). The current study has shown that a significant proportion of children with JMML can be cured with HSCT, especially those receiving the busulfan/fludarabine/melphalan regimen. Based on the lower relapse and better survival observed in patients with chronic GVHD, additional treatment strategies that focus on enhancing graft-versus-leukemia effects may further improve survival.

    DOI: 10.1016/j.bbmt.2019.11.029

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  292. Autologous Stem Cell Transplantation for Children With Renal Tumors, and Adults With Wilms Tumor: Retrospective Analysis of the Japanese Transplant Registry Unified Management Program. Reviewed International journal

    Ryoji Kobayashi, Masami Inoue, Yoshiyuki Takahashi, Atsushi Kikuta, Atsushi Ogawa, Yuhki Koga, Katsuyoshi Koh, Yoshiko Hashii, Junichi Hara, Koji Kato, Ken Tabuchi, Kimikazu Matsumoto

    Journal of pediatric hematology/oncology   Vol. 42 ( 4 ) page: 251 - 255   2020.5

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    BACKGROUND: Almost all pediatric patients with renal tumors are diagnosed with nephroblastoma (Wilms tumor), clear cell sarcoma, or malignant rhabdoid tumor. The choice of treatment is important for relapsed and refractory patients with nephroblastoma. Furthermore, clear cell sarcoma of the kidney (CCSK) and malignant rhabdoid tumor of the kidney (MRTK) have a poor prognosis compared with nephroblastoma. Thus, stem cell transplantation (SCT) is sometimes selected to treat these tumors. PATIENTS AND METHODS: The authors targeted a total of 84 patients with nephroblastoma, CCSK, and MRTK who underwent a first autologous SCT between 1992 and 2014, and were registered in the Japanese Transplant Registry Unified Management Program system. The authors retrospectively analyzed the SCT data for survival rate. RESULTS: Five-year overall survival rates for nephroblastoma, CCSK, and MRTK were 72.4%±6.3%, 46.8%±13.8%, and 36.4%±14.5%, respectively. The event-free survival rates at 5 years were 64.9%±6.7%, 35.7%±12.8%, and 27.3%±13.4%, respectively. The relapse rates at 5 years were 25.3%±11.4%, 46.2%±28.4%, and 60.0%±43.1%, respectively. CONCLUSION: Although the survival rate for nephroblastoma was relatively high, those of CCSK and MRTK were poor.

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  293. ヒト-ヒト感染が疑われる肺Mycobacterium abscessus complex症の小児症例

    稲垣 孝行, 高橋 弘泰, 山田 清文, 八木 哲也, 高橋 義行, 村松 秀城, 手塚 宜行, 安立 なぎさ, 豊留 有香, 長田 ゆかり

    日本化学療法学会雑誌   Vol. 68 ( 3 ) page: 444 - 444   2020.5

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  294. droplet digital PCRを用いた若年性骨髄単球性白血病におけるサブクローナルなSETBP1とJAK3変異の検出

    若松 学, 村上 典寛, 奥野 友介, 北澤 宏展, 山森 彩子, 今屋 雅之, 吉田 太郎, 三輪田 俊介, 成田 幸太郎, 北澤 展典, 濱田 太立, 市川 大輔, 谷口 理恵子, 西川 英里, 川島 希, 成田 敦, 西尾 信博, 村松 秀城, 小島 勢二, 高橋 義行

    日本小児科学会雑誌   Vol. 124 ( 5 ) page: 881 - 881   2020.5

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  295. droplet digital PCRを用いた若年性骨髄単球性白血病におけるサブクローナルなSETBP1とJAK3変異の検出

    若松 学, 村上 典寛, 奥野 友介, 北澤 宏展, 山森 彩子, 今屋 雅之, 吉田 太郎, 三輪田 俊介, 成田 幸太郎, 北澤 展典, 濱田 太立, 市川 大輔, 谷口 理恵子, 西川 英里, 川島 希, 成田 敦, 西尾 信博, 村松 秀城, 小島 勢二, 高橋 義行

    日本小児科学会雑誌   Vol. 124 ( 5 ) page: 881 - 881   2020.5

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  296. Biallelic VPS35L pathogenic variants cause 3C/Ritscher-Schinzel-like syndrome through dysfunction of retriever complex. Reviewed International journal

    Kohji Kato, Yasuyoshi Oka, Hideki Muramatsu, Filipp F Vasilev, Takanobu Otomo, Hisashi Oishi, Yoshihiko Kawano, Hiroyuki Kidokoro, Yuka Nakazawa, Tomoo Ogi, Yoshiyuki Takahashi, Shinji Saitoh

    Journal of medical genetics   Vol. 57 ( 4 ) page: 245 - 253   2020.4

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    BACKGROUND: 3C/Ritscher-Schinzel syndrome is characterised by congenital cranio-cerebello-cardiac dysplasia, where CCDC22 and WASHC5 are accepted as the causative genes. In combination with the retromer or retriever complex, these genes play a role in endosomal membrane protein recycling. We aimed to identify the gene abnormality responsible for the pathogenicity in siblings with a 3C/Ritscher-Schinzel-like syndrome, displaying cranio-cerebello-cardiac dysplasia, coloboma, microphthalmia, chondrodysplasia punctata and complicated skeletal malformation. METHODS: Exome sequencing was performed to identify pathogenic variants. Cellular biological analyses and generation of knockout mice were carried out to elucidate the gene function and pathophysiological significance of the identified variants. RESULTS: We identified compound heterozygous pathogenic variants (c.1097dup; p.Cys366Trpfs*28 and c.2755G>A; p.Ala919Thr) in the VPS35L gene, which encodes a core protein of the retriever complex. The identified missense variant lacked the ability to form the retriever complex, and the frameshift variant induced non-sense-mediated mRNA decay, thereby confirming biallelic loss of function of VPS35L. In addition, VPS35L knockout cells showed decreased autophagic function in nutrient-rich and starvation conditions, as well as following treatment with Torin 1. We also generated Vps35l-/- mice and demonstrated that they were embryonic lethal at an early stage, between E7.5 and E10.5. CONCLUSIONS: Our results suggest that biallelic loss-of-function variants in VPS35L underlies 3C/Ritscher-Schinzel-like syndrome. Furthermore, VPS35L is necessary for autophagic function and essential for early embryonic development. The data presented here provide a new insight into the critical role of the retriever complex in fetal development.

    DOI: 10.1136/jmedgenet-2019-106213

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  297. Mesenchymal stem/stromal cells stably transduced with an inhibitor of CC chemokine ligand 2 ameliorate bronchopulmonary dysplasia and pulmonary hypertension. Reviewed International journal

    Toshihiko Suzuki, Yoshiaki Sato, Hidenori Yamamoto, Taichi Kato, Yuma Kitase, Kazuto Ueda, Haruka Mimatsu, Yuichiro Sugiyama, Atsuto Onoda, Shigeki Saito, Yoshiyuki Takahashi, Takayuki Nakayama, Masahiro Hayakawa

    Cytotherapy   Vol. 22 ( 4 ) page: 180 - 192   2020.4

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    Perinatal bronchopulmonary dysplasia (BPD) is defined as lung injury in preterm infants caused by various factors, resulting in serious respiratory dysfunction and high mortality. The administration of mesenchymal stem/stromal cells (MSCs) to treat/prevent BPD has proven to have certain therapeutic effects. However, MSCs can only weakly regulate macrophage function, which is strongly involved in the development of BPD. 7ND-MSCs are MSCs transfected with 7ND, a truncated version of CC chemokine ligand 2 (CCL2) that promotes macrophage activation, using a lentiviral vector. In the present study, we show in a BPD rat model that 7ND-MSC administration, but not MSCs alone, ameliorated the impaired alveolarization evaluated by volume density and surface area in the lung tissue, as well as pulmonary artery remodeling and pulmonary hypertension induced by BPD. In addition, 7ND-MSCs, but not MSCs alone, reduced M1 macrophages and the messenger RNA expressions of interleukin-6 and CCL2 in the lung tissue. Thus, the present study showed the treatment effect of 7ND-MSCs in a BPD rat model, which was more effective than that of MSCs alone.

    DOI: 10.1016/j.jcyt.2020.01.009

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  298. Clinical Characteristics and Treatment Outcomes of Pediatric Patients with Non-Hodgkin Lymphoma in East Asia. Reviewed International journal

    Jin Kyung Suh, Yi-Jin Gao, Jing-Yan Tang, Shiann-Tarng Jou, Dong-Tsamn Lin, Yoshiyuki Takahashi, Seiji Kojima, Ling Jin, Yonghong Zhang, Jong Jin Seo

    Cancer research and treatment : official journal of Korean Cancer Association   Vol. 52 ( 2 ) page: 359 - 368   2020.4

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    PURPOSE: The presentations and geographic incidence of pediatric non-Hodgkin lymphoma (NHL) differ from those of adults. This study delineated the characteristics and outcomes of pediatric NHL in East Asia. MATERIALS AND METHODS: Medical records of 749 pediatric patients with NHL treated at participating institutions in mainland China, Japan, Korea, and Taiwan from January 2008 to December 2013 were reviewed. Demographic and clinical features, survival outcomes, and putative prognostic factors were analyzed. RESULTS: Five hundred thirty patients (71%) were male. The most common pathologic subtypes were Burkitt lymphoma (BL) (36%). Six hundred seven patients (81%) had advanced diseases at diagnosis. The 5-year overall survival and event-free survival (EFS) rates were 89% and 84%. The 5-year EFS rates of BL, lymphoblastic lymphoma, and diffuse large B-cell lymphoma were 88%, 88%, and 89%, and those of anaplastic large cell lymphoma (ALCL) and peripheral T-cell lymphoma (PTCL) were 71% and 56% (p < 0.001). Central nervous system involvement, high lactate dehydrogenase level (> 250 IU/mL), and advanced disease at diagnosis (≥ stage III) were associated with poor outcomes (p < 0.05). ALCL and PTCL relapsed more frequently than other pathologic subtypes (p < 0.001). CONCLUSION: In East Asia, PTCL was more frequent than in Western countries, and bone marrow involvement did not affect treatment outcome. This international study should motivate future collaborative study on NHL in East Asia.

    DOI: 10.4143/crt.2019.219

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  299. 【小児血液・腫瘍疾患の最前線】診療のトピックス 高リスク神経芽腫に対するGD2抗体療法

    高橋 義行

    小児科診療   Vol. 83 ( 4 ) page: 455 - 458   2020.4

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    ●神経芽腫に対する免疫療法の標的分子として、ジシアロガングリオシドGD2抗原タンパクに対するモノクローナル抗体がGD2抗体である。●治療毒性(疼痛、アレルギー反応、血管漏出症候群など)があるものの管理は可能で、欧米ではすでに高リスク神経芽腫の標準治療薬として保険承認されている。●わが国では米国製剤、欧州製剤ともに治験が行われており、ドラッグラグ解消のため早期承認が望まれている。(著者抄録)

  300. Novel compound heterozygous MCOLN1 mutations identified in a Japanese girl with severe developmental delay and thin corpus callosum. Reviewed International journal

    Naoya Yamaguchi, Kyoko Ban, Atsushi Suzuki, Yuji Nakamura, Kohji Kato, Hideki Muramatsu, Yusuke Okuno, Ayako Hattori, Tadashi Kaname, Yoshiyuki Takahashi, Shinji Saitoh

    Brain & development   Vol. 42 ( 3 ) page: 298 - 301   2020.3

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    Mucolipidosis type IV (MLIV) is a rare lysosomal storage disorder causing severe psychomotor developmental delay and progressive visual impairment. MLIV is an autosomal recessive disease caused by mutations in MCOLN1, which encodes for mucolipin-1. Here, we report a case of a 4-year-old Japanese girl with severe intellectual disability and motor deficits. Brain magnetic resonance imaging showed signal abnormalities in the white matter and thinning of the corpus callosum. Whole-exome sequencing was performed on the proband and her parents, and novel compound heterozygous mutations at c.936_938del (p.Phe313del) and c.1503dupC (p.Ile502Hisfs*106) in MCOLN1 (NM_020533.2) were identified in the proband. Additional biochemical examinations revealed elevated serum gastrin level and iron deficiency anemia, leading to the diagnosis of MLIV. More reports of such pathogenic mutations are expected to broaden the understanding of the channel function of mucolipin-1 and genotype-phenotype correlations.

    DOI: 10.1016/j.braindev.2019.12.003

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  301. Respiratory illness and acute flaccid myelitis in the Tokai district in 2018. Reviewed International journal

    Akihisa Okumura, Shingo Numoto, Hideyuki Iwayama, Hirokazu Kurahashi, Jun Natsume, Shinji Saitoh, Tetsushi Yoshikawa, Toshiyuki Fukao, Masahiro Hirayama, Yoshiyuki Takahashi

    Pediatrics international : official journal of the Japan Pediatric Society   Vol. 62 ( 3 ) page: 337 - 340   2020.3

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    BACKGROUND: An outbreak of acute flaccid myelitis was chronologically correlated with an outbreak of severe respiratory illness in Japan in 2015. We hypothesized that increases in children hospitalized with severe respiratory illnesses might also be associated with increase in acute flaccid myelitis in autumn 2018. METHODS: We explored the temporal correlations between respiratory illness outbreaks and acute flaccid myelitis during autumn season between 2016 and 2018 using questionnaire surveys. One questionnaire explored the monthly numbers of children with acute flaccid myelitis, Guillain-Barré syndrome, and other acute flaccid paralyses. The other questionnaire explored the monthly numbers of children hospitalized with respiratory illnesses associated with wheezing. A correlation between the monthly numbers of children with acute flaccid myelitis and those with respiratory illness was analyzed using the Pearson correlation test. RESULTS: Although the number of patients hospitalized with respiratory illness did not correlate with the number of those admitted with myelitis, increases in children aged 7-12 and 13-19 years requiring intensive care unit admission correlated temporally with an outbreak of acute flaccid myelitis. CONCLUSIONS: An increase in intensive care unit admissions to treat respiratory disease occurred in association with a cluster of acute flaccid myelitis. An increase in the number of intensive care unit admissions due to respiratory illness may be a clue to expect the occurrence of acute flaccid myelitis.

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  302. 繰り返す胸痛の経過中に突然発症した心室頻拍の1例

    三輪 剛士, 山本 英範, 深澤 佳絵, 六鹿 雅登, 加藤 太一, 高橋 義行

    日本小児科学会雑誌   Vol. 124 ( 3 ) page: 611 - 612   2020.3

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  303. 早期よりエトレチナートを投与し効果がみられた道化師様魚鱗癬の1例

    近藤 真生, 浅田 英之, 本部 和也, 郷間 環, 新井 紗記子, 上田 一仁, 三浦 良介, 呉 尚治, 兵藤 玲奈, 鈴木 俊彦, 伊藤 美春, 齊藤 明子, 村松 友佳子, 佐藤 義朗, 山本 ひかる, 加藤 有一, 杉山 裕一郎, 高橋 義行, 早川 昌弘

    日本小児科学会雑誌   Vol. 124 ( 3 ) page: 609 - 610   2020.3

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  304. 急性弛緩性脊髄炎による左上肢麻痺に対して神経移行術を施行した1例

    西尾 洋介, 中田 智彦, 白木 杏奈, 鈴木 健史, 川口 将宏, 牧 祐輝, 田中 雅大, 坂口 陽子, 成田 敦, 山本 啓之, 城所 博之, 梶田 光春, 平田 仁, 高橋 義行, 夏目 淳

    日本小児科学会雑誌   Vol. 124 ( 3 ) page: 604 - 604   2020.3

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  305. 小児がん・白血病治療における最新医療 次世代シークエンサーの臨床応用と遺伝子導入T細胞療法

    高橋 義行

    日本小児科学会雑誌   Vol. 124 ( 3 ) page: 582 - 582   2020.3

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  306. TREC/KRECキットを用いた新生児マススクリーニング検査法の有用性

    若松 学, 村松 秀城, 片岡 伸介, 山森 彩子, 今屋 雅之, 吉田 太郎, 三輪田 俊介, 成田 幸太郎, 北澤 宏展, 濱田 太立, 市川 大輔, 谷口 理恵子, 西川 英里, 川島 希, 成田 敦, 奥野 友介, 西尾 信博, 中島 葉子, 伊藤 哲哉, 柘植 郁哉, 中村 富美子, 酒井 好美, 小島 勢二, 高橋 義行

    日本小児科学会雑誌   Vol. 124 ( 3 ) page: 605 - 605   2020.3

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  307. Multilineage-differentiating stress enduring cellsの新生児低酸素性虚血性脳症に対する治療効果の検討

    鈴木 俊彦, 佐藤 義朗, 上田 一仁, 北瀬 悠磨, 呉 尚治, 三浦 良介, 浅田 英之, 兵藤 玲奈, 伊藤 美春, 齊藤 明子, 村松 友佳子, 高橋 義行, 早川 昌弘

    日本小児科学会雑誌   Vol. 124 ( 3 ) page: 595 - 596   2020.3

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  308. Human herpesvirus-6B infection in pediatric allogenic hematopoietic stem cell transplant patients: Risk factors and encephalitis. Reviewed International journal

    Hiroki Miura, Yoshiki Kawamura, Fumihiko Hattori, Makito Tanaka, Kazuko Kudo, Masaru Ihira, Hiroshi Yatsuya, Yoshiyuki Takahashi, Seiji Kojima, Hirotoshi Sakaguchi, Nao Yoshida, Asahito Hama, Tetsushi Yoshikawa

    Transplant infectious disease : an official journal of the Transplantation Society   Vol. 22 ( 1 ) page: e13203   2020.2

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    BACKGROUND: Human herpesvirus-6B (HHV-6B) infection after allogenic hematopoietic stem cell transplantation (allo-HSCT) is known to be associated with post-transplant limbic encephalitis in adults. Meanwhile, the association between HHV-6B infection and central nervous system complications remains unclear in pediatric allo-HSCT patients. METHODS: In this study, HHV-6B infection was monitored for more than 50 days after HSCT using virus isolation and real-time PCR. Clinical information such as patient background and encephalitis status was collected retrospectively from medical records. Risk factors for HHV-6B infection were determined by the Cox proportional hazards model, and the clinical features of HHV-6B encephalitis in pediatric allo-HSCT patients were elucidated. RESULTS: Human herpesvirus-6B infection was observed in 74 (33.8%) of 219 patients at 3-47 days (median 18, interquartile range 13-20). Risk factors identified in multivariable analysis were hematological malignancy (hazards ratio [HR], 5.0; 95% confidence interval [CI], 2.3/12.5; P < .0001), solid tumor (HR, 4.8; CI, 1.5/16.3; P = .0104), unrelated donor (HR, 2.1; CI, 1.0/4.6; P = .0378), and sex-mismatched donor (HR 1.8; CI, 1.1/3.0; P = .0257). HHV-6B encephalitis occurred in only one of the 219 patients (0.46%); this patient demonstrated the typical clinical course of posterior reversible encephalopathy syndrome. CONCLUSION: Hematological malignancy, solid tumor, unrelated donor, and sex-mismatched donor were significant risk factors for HHV-6B infection after pediatric allo-HSCT. In pediatric allo-HSCT patients, the incidence of HHV-6B encephalitis was low and the clinical features differed from those in adult patients.

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  309. Pseudo-sawtooth pattern on amplitude-integrated electroencephalography in neonatal hypoxic-ischemic encephalopathy. International journal

    Masaharu Tanaka, Hiroyuki Kidokoro, Tetsuo Kubota, Tatsuya Fukasawa, Yu Okai, Yoko Sakaguchi, Yuji Ito, Hiroyuki Yamamoto, Atsuko Ohno, Tomohiko Nakata, Tamiko Negoro, Akihisa Okumura, Toru Kato, Kazuyoshi Watanabe, Yoshiyuki Takahashi, Jun Natsume

    Pediatric research   Vol. 87 ( 3 ) page: 529 - 535   2020.2

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    OBJECTIVE: The objective of this study was to describe a novel amplitude-integrated electroencephalography (aEEG) pattern in infants with hypoxic-ischemic encephalopathy (HIE) and to assess the clinical significance. METHODS: The aEEG traces of infants with HIE who were treated with therapeutic hypothermia (TH) from 2012 to 2017 were analyzed. A pseudo-sawtooth (PST) pattern was defined as a periodic increase of the upper and/or lower margin of the trace on aEEG without showing seizure activities on conventional EEG (CEEG). RESULTS: Of the 46 infants, 6 (13%) had the PST pattern. The PST pattern appeared following a flat trace or a continuous low-voltage pattern and was followed by a burst-suppression pattern. On CEEG, the PST pattern consists of alternating cycles of low-voltage irregular activities and almost flat tracing. The PST pattern was associated with neuroimaging abnormalities and with various degrees of neurodevelopmental outcomes. Positive predictive values of the PST or worse pattern for adverse outcomes were high at 12 h after birth. CONCLUSION: A novel aEEG background pattern in infants with HIE was reported. The PST pattern likely indicates a suppressed background pattern and may be linked to unfavorable outcomes. Further multicenter validation study is needed to clarify its clinical significance.

    DOI: 10.1038/s41390-019-0567-5

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  310. 造血幹細胞移植を受けた小児における拡散テンソル画像解析

    坂口 陽子, 城所 博之, 白木 杏奈, 鈴木 健史, 川口 将宏, 牧 祐輝, 田中 雅大, 山本 啓之, 中田 智彦, 村松 秀城, 高橋 義行, 夏目 淳

    日本小児科学会雑誌   Vol. 124 ( 2 ) page: 290 - 290   2020.2

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  311. 神経芽腫寛解導入療法における重症呼吸器症状のリスク因子の検討(Severe respiratory symptoms during induction chemotherapy in neuroblastoma)

    松井 基浩, 山岡 祥子, 斎藤 雄弥, 横川 裕一, 牧本 敦, 西川 英里, 川島 希, 成田 敦, 西尾 信博, 村松 秀城, 高橋 義行, 浦島 充佳, 湯坐 有希

    日本小児科学会雑誌   Vol. 124 ( 2 ) page: 294 - 294   2020.2

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  312. 腫瘍性疾患と鑑別を要したPhlegmonous gastritisの小児例

    三輪田 俊介, 成田 敦, 吉田 太郎, 今屋 雅之, 山森 彩子, 若松 学, 成田 幸太郎, 北澤 宏展, 濱田 太立, 市川 大輔, 谷口 理恵子, 西川 英里, 川島 希, 奥野 友介, 村松 秀城, 西尾 信博, 小島 勢二, 阿部 直紀, 岩田 直美, 高橋 義行

    日本小児科学会雑誌   Vol. 124 ( 2 ) page: 333 - 333   2020.2

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  313. 川崎病症例における血清中微生物メタゲノム解析

    鳥居 ゆか, 堀場 千尋, 早野 聡, 加藤 太一, 鈴木 高子, 川田 潤一, 高橋 義行, 小島 勢二, 伊藤 嘉規

    日本小児科学会雑誌   Vol. 124 ( 2 ) page: 223 - 223   2020.2

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  314. Targetable driver mutations in multicentric reticulohistiocytosis. Reviewed International journal

    Norihiro Murakami, Tomohisa Sakai, Eisuke Arai, Hideki Muramatsu, Daisuke Ichikawa, Shuji Asai, Yoshie Shimoyama, Naoki Ishiguro, Yoshiyuki Takahashi, Yusuke Okuno, Yoshihiro Nishida

    Haematologica   Vol. 105 ( 2 ) page: E61 - E64   2020.1

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    DOI: 10.3324/haematol.2019.218735

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  315. A Novel Treatment with Stem Cells from Human Exfoliated Deciduous Teeth for Hypoxic-Ischemic Encephalopathy in Neonatal Rats. Reviewed International journal

    Yuma Kitase, Yoshiaki Sato, Kazuto Ueda, Toshihiko Suzuki, Alkisti Mikrogeorgiou, Yuichiro Sugiyama, Kohki Matsubara, Yuka Tsukagoshi Okabe, Shinobu Shimizu, Hitoshi Hirata, Hiroshi Yukawa, Yoshinobu Baba, Masahiro Tsuji, Yoshiyuki Takahashi, Akihito Yamamoto, Masahiro Hayakawa

    Stem cells and development   Vol. 29 ( 2 ) page: 63 - 74   2020.1

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    Recently, cell therapy has been developed as a novel treatment for perinatal hypoxic-ischemic encephalopathy (HIE), which is an important cause of neurological disorder and death, and stem cells from human exfoliated deciduous teeth (SHED) express early markers for mesenchymal and neuroectodermal stem cells. We investigated the treatment effect of SHED for HIE in neonatal rats. Seven-day-old rats underwent ligation of the left carotid artery and were exposed to 8% hypoxic treatment. SHED (1 × 105 cells) were injected via the right external jugular vein 24 h after the insult. The effect of intravenous administration of SHED cells was evaluated neurologically and pathophysiologically. In the evaluation of engraftment using quantum dots 655, only a few SHED were detected in the injured cortex. In the immunohistological evaluation 24 h after injection, the numbers of positive cells of active caspase-3 and anti-4 hydroxynonenal antiserum were lower in the SHED group than in the vehicle group. The number of Iba-1+ cells in the cortex was higher in the SHED group. However, the proportion of M1 microglia (Iba-1+/ED-1+) was significantly decreased, whereas M2 microglia (Iba-1+/CD206+) tended to increase in the SHED group. In the behavioral tests performed 5 months after hypoxic treatment, compared to the vehicle group, the SHED group showed significant elongation of the endurance time in the rotarod treadmill test, significantly ameliorated proportion of using the impaired hand in the cylinder test, significantly lower ratio of right/left front paw area in gait analysis, and significantly higher avoidance rate in the active avoidance test. In the in vitro experiment with cultured neurons exposed to oxygen-glucose deprivation, we confirmed the neuroprotective effect of the condition medium of SHED. These results suggested that intravenous administration of SHED exerted a treatment effect both histologically and functionally, possibly via a paracrine effect.

    DOI: 10.1089/scd.2019.0221

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  316. A Case of a 14-Year-Old Boy who had Difficulty Walking Due to Drug-Induced Neuropathy During T-Cell Acute Lymphocytic Leukemia Treatment

    Taro Yoshida, Aya Sawamura, Hideki Muramatsu, Masayuki Imaya, Ayako Yamamori, Manabu Wakamatsu, Shunsuke Miwata, Kotaro Narita, Hironobu Kitazawa, Rieko Taniguchi, Daisuke Ichikawa, Eri Nishikawa, Motoharu Hamada, Nozomu Kawashima, Atsushi Narita, Nobuhiro Nishio, Seiji Kojima, Tomohiko Nakata, Hiroyuki Kidokoro, Jun Natxume, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S30 - S30   2019.12

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  317. A CaseWith Dyskeratosis Congenita Achieved Transfusion Independence after Eltrombopag Treatment

    Wakamatsu, M; Muramatsu, H; Imaya, M; Yamamori, A; Yoshida, T; Miwata, S; Kitazawa, H; Narita, K; Ichikawa, D; Hamada, M; Taniguchi, R; Kawasima, N; Nishikawa, E; Narita, A; Okuno, Y; Nishio, N; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S46 - S46   2019.12

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  318. Clinical and Laboratory Characteristics of Atypical Chronic Myeloid Leukemia and Chronic Myelomonocytic Leukemia in Japanese Children

    Hama, A; Ito, M; Hasegawa, D; Nozawa, K; Muramatsu, H; Takahashi, Y; Watanabe, K; Ohara, A; Manabe, A; Kojima, S

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S21 - S22   2019.12

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  319. An Infant With Hereditary Folate Malabsorption Requiring Differentiation from Acute Megakaryoblastic Leukemia

    Eri Nishikawa, Rieko Taniguchi, Tarou Yoshida, Masayuki Imaya, Manabu Wakamatsu, Syunsuke Miwata, Koutarou Narita, Ayako Yamamori, Hironobu Kitazawa, Daisuke Ichikawa, Motoharu Hamada, Nozomu Kawashima, Atsushi Narita, Yusuke Okuno, Hideki Muramatsu, Nobuhiro Nishio, Seiji Kojima, Shimaa Said, Yusuke Tozawa, Masafumi Yamada, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S73 - S73   2019.12

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  320. Identification of <i>FANCI</i> Mutation in Fanconi Anemia in Infancy

    Urata, T; Imamura, T; Osone, S; Muramatsu, H; Takahashi, Y; Hosoi, H

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S44 - S44   2019.12

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  321. Induction of Partial Response With Ceritinib for a Child With Chemotherapy-Refractory ALK-rearranged Neuroblastoma

    Kotaro Narita, Taro Yoshida, Ayako Yamamori, Manabu Wakamatsu, Masayuki Imaya, Shunsuke Miwata, Hironobu Kitazawa, Rieko Taniguchi, Daisuke Ichikawa, Motoharu Hamada, Eri Nishikawa, Nobuhiro Nishio, Yusuke Okuno, Nozomu Kawashima, Atsushi Narita, Hideki Muramatsu, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S104 - S104   2019.12

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  322. Correlation Between Plasma Rabbit Anti-Thymocyte Globulin Level and Response to Immunosuppressive Therapy in Patients With Severe Aplastic Anemia

    Atsushi Narita, Hideki Muramatsu, Motoharu Hamada, Daisuke Ichikawa, Eri Nishikawa, Nozomu Kawashima, Yusuke Okuno, Nobuhiro Nishio, Hirohito Yamazaki, Shinji Nakao, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S45 - S45   2019.12

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  323. Conditioning Regimen for Allogeneic Bone Marrow Transplantation in Children With Acquired Bone Marrow Failure: Fludarabine/Melphalan vs. Fludarabine/Cyclophosphamide

    Yoshida, N; Takahashi, Y; Yabe, H; Kobayashi, R; Watanabe, K; Kudo, K; Yabe, M; Miyamura, T; Koh, K; Kawaguchi, H; Goto, H; Fujita, N; Hara, J; Okamoto, Y; Kato, K; Inoue, M; Suzuki, R; Atsuta, Y; Kojima, S

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S95 - S95   2019.12

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  324. Integrating FANCD2 Functional Analysis With High-Throughput Sequencing for Diagnosing Fanconi Anemia

    Nozomu Kawashima, Yusuke Okuno, Hideki Muramatsu, Rieko Taniguchi, Taro Yoshida, Masayuki Imaya, Ayako Yamamori, Manabu Wakamatsu, Shunsuke Miwata, Kotaro Narita, Motoharu Hamada, Daisuke Ichikawa, Nobuhiro Kitazawa, Eri Nishikawa, Atsushi Narita, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S44 - S45   2019.12

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  325. Evaluation of the Outcome of KIR-Ligand Mismatched Cord Blood Transplantation for High Risk Metastatic Neuroblastoma Using New SIOPEN Risk Stratification

    Nobuhiro Nishio, Daisuke Ichikawa, Manabu Wakamatsu, Kotaro Narita, Shunsuke Miwata, Hironobu Kitazawa, Rieko Taniguchi, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Yusuke Okuno, Atsushi Narita, Hideki Muramatsu, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S106 - S106   2019.12

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  326. Genetic Diagnosis Based on a Comprehensive Fusion Gene Analysis of Pediatric Malignant Solid Tumors

    Daisuke Ichikawa, Hideki Muramatsu, Yusuke Okuno, Motoharu Hamada, Rieko Taniguchi, Eri Nishikawa, Nozomu Kawashima, Atsushi Narita, Nobuhiro Nishio, Yoshie Shimoyama, Masato Nakaguro, Shigeo Nakamura, Seiji Kojima, Atsuko Nakazawa, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S61 - S61   2019.12

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  327. Two Cases Effectively Treated with Oral Budesonide Administration for Steroid-resistant Intestinal Acute Graft Versus Host Disease

    Ayako Yamamori, Eri Nishikawa, Atsushi Narita, Masayuki Imaya, Manabu Wakamatsu, Taro Yoshida, Shunsuke Miwata, Hironobu Kitazawa, Kotaro Narita, Rieko Taniguchi, Daisuke Ichikawa, Motoharu Hamada, Nozomu Kawashima, Yusuke Okuno, Nobuhiro Nishio, Hideki Muramatsu, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S37 - S38   2019.12

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  328. Whole Exome Sequencing Contributes to Genetic Diagnosis of IBMFS Patients

    Hideki Muramatsu, Motoharu Hamada, Yusuke Okuno, Manabu Wakamatsu, Rieko Taniguchi, Koutarou Narita, Nozomu Kawashima, Hironobu Kitazawa, Daisuke Ichikawa, Eri Nishikawa, Nazomu Kawashima, Atsushi Narita, Nobuhiro Nishio, Seiji Kojima, Tomoo Ogi, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S63 - S64   2019.12

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  329. Prediction of postnatal clinical course in primary congenital dilated cardiomyopathy. Reviewed International journal

    Hidenori Yamamoto, Yoshie Fukasawa, Naoki Ohashi, Takehiko Yokoyama, Kazutaka Suzuki, Takaya Ota, Kazushi Yasuda, Kentaro Omoya, Yoshiyuki Takahashi, Taichi Kato

    Pediatrics international : official journal of the Japan Pediatric Society   Vol. 61 ( 12 ) page: 1196 - 1201   2019.12

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    BACKGROUND: The aim of this study was to investigate the prediction of postnatal prognosis using fetal and perinatal data in patients with primary congenital dilated cardiomyopathy (PCDCM), and to estimate the incidence of this disease. METHODS: We examined correlations between fetal or perinatal data and postnatal clinical course in a multicenter retrospective study of eight patients with PCDCM. Incidence was calculated in a population-based study. RESULTS: All patients developed heart failure at a median of 8 days (range, 0-43 days), and six patients died or required extracorporeal artificial heart therapy at a median of 67 days (range, 0-92 days). The cardiothoracic area ratio from fetal echocardiography, the Apgar score, and the standard deviation of birth weight correlated significantly with the date at onset of heart failure. However, no data correlated with survival. Cumulative incidence of PCDCM was calculated as 1.21 per 100 000 total births (95% confidence interval, 0.37 to 2.06). CONCLUSIONS: Primary congenital dilated cardiomyopathy has a poor prognosis, but cardiothoracic area ratio from fetal echocardiography, body weight at birth, and Apgar score correlate with the timing of the onset of heart failure, and these indicators might therefore be useful for peri- and postnatal management.

    DOI: 10.1111/ped.14029

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  330. Successful Treatment of Central Nervous System Relapses of Neuroblastoma Stage 4 After a KIR-ligand Mismatched Allogeneic Cord Blood Transplantation

    Masayuki Imaya, Atsushi Narita, Taro Yoshida, Ayako Yamamori, Manabu Wakamatsu, Syunsuke Miwata, Kotaro Narita, Hironobu Kitazawa, Rieko Taniguchi, Daisuke Ichikawa, Motoharu Hamada, Eri Nishikawa, Nozomu Kawashima, Hideki Muramatsu, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S102 - S102   2019.12

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  331. Successfully Treated Three Children With Pericardial Effusion After Hematopoietic Stem Cell Transplantation

    Hironobu Kitazawa, Atsushi Narita, Taro Yoshida, Masayuki Imaya, Ayako Yamamori, Manabu Wakamatsu, Shunsuke Miwata, Kotaro Narita, Rieko Taniguchi, Motoharu Hamada, Daisuke Ichikawa, Eri Nishikawa, Nozomu Kawashima, Yusuke Okuno, Nobuhiro Nishio, Hideki Muramatsu, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 66   page: S97 - S97   2019.12

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  332. Comprehensive Mutational Analysis of Juvenile Myelomonocytic Leukemia Using Whole-Genome Sequencing

    Yusuke Okuno, Hideki Muramatsu, Norihiro Murakami, Nozomu Kawashima, Manabu Wakamatsu, Hironobu Kitazawa, Tomoo Ogi, Yoshiyuki Takahashi

    BLOOD   Vol. 134   2019.11

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    DOI: 10.1182/blood-2019-121681

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  333. DNA Methylation As a Biomarker of Outcome in JMML: An International Effort Towards Clinical Implementation

    Meyer, J; Schönung, M; Flotho, C; Olshen, AB; Hartmann, M; Plass, C; Okuno, Y; Takahashi, Y; Murakami, N; Wakamatsu, M; Loh, ML; Niemeyer, CM; Muramatsu, H; Lipka, DB; Stieglitz, E

    BLOOD   Vol. 134   2019.11

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    DOI: 10.1182/blood-2019-131279

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  334. Detection of Subclonal <i>SETBP1</i> and <i>JAK3</i> Mutations in Patients with Juvenile Myelomonocytic Leukemia Using Droplet Digital PCR

    Wakamatsu, M; Muramatsu, H; Murakami, N; Okuno, Y; Kitazawa, H; Kojima, S; Takahashi, Y

    BLOOD   Vol. 134   2019.11

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    DOI: 10.1182/blood-2019-125354

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  335. Genome-Wide Methylation Analysis Using the Digital Restriction Enzyme Analysis of Methylation for Stratification of Patients with Juvenile Myelomonocytic Leukemia

    Hironobu Kitazawa, Hideki Muramatsu, Norihiro Murakami, Yusuke Okuno, Manabu Wakamatsu, Taro Yoshida, Masayuki Imaya, Ayako Yamamori, Shunsuke Miwata, Kotaro Narita, Motoharu Hamada, Daisuke Ichikawa, Rieko Taniguchi, Nozomu Kawashima, Eri Nishikawa, Atsushi Narita, Nobuhiro Nishio, Seiji Kojima, Yoshiyuki Takahashi

    BLOOD   Vol. 134   2019.11

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    DOI: 10.1182/blood-2019-127792

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  336. High Natural Killer Cell Count at Diagnosis Predicts a Good Response to Immunosuppressive Therapy in Aplastic Anemia

    Atsushi Narita, Hideki Muramatsu, Yinyan Xu, Motoharu Hamada, Daisuke Ichikawa, Eri Nishikawa, Nozomu Kawashima, Yusuke Okuno, Nobuhiro Nishio, Hirohito Yamazaki, Shinji Nakao, Seiji Kojima, Yoshiyuki Takahashi

    BLOOD   Vol. 134   2019.11

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    DOI: 10.1182/blood-2019-124945

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  337. Utility of Newborn Screening for Severe Combined Immunodeficiency and X-Linked Agammaglobulinemia Using TREC and KREC Assays

    Wakamatsu, M; Muramatsu, H; Kataoka, S; Okuno, Y; Yoshimi, S; Nakajima, Y; Ito, T; Tsuge, I; Kojima, S; Takahashi, Y

    BLOOD   Vol. 134   2019.11

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    DOI: 10.1182/blood-2019-126669

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  338. Phase I Study of Cord Blood Transplantation with Intra-Bone Marrow Injection of Mesenchymal Stem Cells

    Goto, T; Murata, M; Nishida, T; Terakura, S; Kamoshita, S; Ishikawa, Y; Ushijima, Y; Adachi, Y; Kato, K; Hirakawa, A; Nishiwaki, S; Nishio, N; Takahashi, Y; Kodera, Y; Matsushita, T; Kiyoi, H

    BLOOD   Vol. 134   2019.11

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    DOI: 10.1182/blood-2019-122101

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  339. Corrigendum to "Frequent intragenic microdeletions of elastin in familial supravalvular aortic stenosis." [Int. J. Cardiol., 274 (2019) 290-295]. Reviewed International journal

    Satoshi Hayano, Yusuke Okuno, Makiko Tsutsumi, Hidehito Inagaki, Yoshie Fukasawa, Hiroki Kurahashi, Seiji Kojima, Yoshiyuki Takahashi, Taichi Kato

    International journal of cardiology   Vol. 292   page: 283 - 283   2019.10

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    DOI: 10.1016/j.ijcard.2019.06.020

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  340. Prospective randomized trial comparing two doses of rabbit anti-thymocyte globulin in patients with severe aplastic anaemia. Reviewed International journal

    Atsushi Narita, Xiaofan Zhu, Hideki Muramatsu, Xiaojuan Chen, Ye Guo, Wenyu Yang, Jingliao Zhang, Fang Liu, Jun H Jang, Hoon Kook, Hawk Kim, Kensuke Usuki, Hirohito Yamazaki, Yoshiyuki Takahashi, Shinji Nakao, Jong Wook Lee, Seiji Kojima

    British journal of haematology   Vol. 187 ( 2 ) page: 227 - 237   2019.10

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    The treatment of choice for patients with severe aplastic anaemia (SAA) includes immunosuppressive therapy (IST) with anti-thymocyte globulin (ATG) and ciclosporin A. However, the optimal dose for rabbit ATG has yet to be established. We herein report the first prospective, randomized, multicentre study comparing two doses of rabbit ATG in patients with SAA. Patients with SAA who required initial IST in Japan (n = 89), China (n = 85) and Korea (n = 48) were enrolled between May 2012 and October 2017. A 1:1 block randomization was employed for two doses of rabbit ATG. In total, 222 patients were randomized, with 112 patients receiving 2·5 mg/kg and 110 receiving 3·5 mg/kg of rabbit ATG for 5 days. The primary endpoint was the haematological response at day 180. After 6 months, no significant difference in response rates was observed between the 2·5 and 3·5 mg/kg groups (49% vs. 48%, P = 0·894). Overall survival at 3 years was similar between the two groups [85% (95% confidence interval [CI], 76%-91%) vs. 91% (95% CI, 82%-96%); P = 0·107]. The current study revealed no significant differences in the efficacy and safety between the 2·5 and 3·5 mg/kg doses of rabbit ATG in patients with SAA. Trial registration: UMIN000011134.

    DOI: 10.1111/bjh.16055

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  341. Retrospective analysis of children with high-risk acute myeloid leukemia who underwent allogeneic hematopoietic stem cell transplantation following complete remission with initial induction chemotherapy in the AML-05 clinical trial. Reviewed International journal

    Nobuyuki Hyakuna, Yoshiko Hashii, Hiroyuki Ishida, Katsutsugu Umeda, Yoshiyuki Takahashi, Masayuki Nagasawa, Hiromasa Yabe, Yozo Nakazawa, Katsuyoshi Koh, Hiroaki Goto, Hiroyuki Fujisaki, Kimikazu Matsumoto, Harumi Kakuda, Michihiro Yano, Akio Tawa, Daisuke Tomizawa, Takashi Taga, Souichi Adachi, Koji Kato

    Pediatric blood & cancer   Vol. 66 ( 10 ) page: e27875   2019.10

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    In the AML-05 clinical trial conducted by the Japanese Pediatric Leukemia/Lymphoma Group from 2006 to 2010, children with high-risk acute myeloid leukemia (HR AML) received allogeneic hematopoietic stem cell transplantation (allo-HSCT) at first complete remission (CR1). The aim of this study was to investigate the impact of allo-HSCT on the outcome of HR AML. Patients with either monosomy 7, 5q-, t(16;21), Ph1, FLT3-ITD, or induction failure after the first course of chemotherapy were eligible for transplant. Of 53 children with HR AML, 51 received allo-HSCT-45 in CR1, five in CR2, and one with non-CR. t(8;21), t(9;11), and t(16;21) abnormalities were identified in eight, five, and four patients, respectively. The stem cell sources varied-bone marrow in 30 patients, peripheral blood in three, and cord blood in 18. The median follow-up was 62 months. The overall survival (OS) rates at 3 years were 73% and 25% for patients who received transplant at CR1 and ≥CR2, respectively. Multivariable analysis showed that patients with chronic graft-versus-host disease (cGVHD) had better OS. This study supports that allo-HSCT is a suitable treatment for HR AML in CR1. The favorable outcome associated with cGVHD indicates that a graft-versus-leukemia effect might be occurring.

    DOI: 10.1002/pbc.27875

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  342. 重症複合型免疫不全患児から排泄されたRV1ワクチン株の院内伝播

    三浦 浩樹, 河村 吉紀, 小澤 慶, 成田 幸太郎, 村松 秀城, 高橋 義行, 安岡 竜平, 井平 勝, 吉川 哲史

    日本小児感染症学会総会・学術集会プログラム・抄録集   Vol. 51回   page: 195 - 195   2019.10

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  343. 日本のAYA世代のがん対策の方向性を読み解く

    栗本 景介, 堀部 敬三, 園原 史訓, 猪川 祥邦, 高見 秀樹, 林 真路, 神田 光郎, 小林 大介, 田中 千恵, 山田 豪, 中山 吾郎, 小池 聖彦, 清水 千佳子, 高橋 義行, 小寺 泰弘

    日本癌治療学会学術集会抄録集   Vol. 57回   page: P166 - 5   2019.10

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  344. 最期の時間を在宅で過ごすことを選んだ高校生の一例 予後告知前後の発言に焦点を当てて

    佐々木 美和, 滝 良梨子, 牧田 夏美, 萩原 沙織, 清水 直子, 梶山 早苗, 谷口 理恵子, 村松 秀城, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 375 - 375   2019.10

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  345. 造血幹細胞移植後に心嚢液貯留をきたした3例(Successfully treated three children with pericardial effusion after hematopoietic stem cell transplantation)

    Kitazawa Hironobu, Narita Atsushi, Yoshida Taro, Imaya Masayuki, Yamamori Ayako, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Taniguchi Rieko, Hamada Motoharu, Ichikawa Daisuke, Nishikawa Eri, Kawashima Nozomu, Okuno Yusuke, Nishio Nobuhiro, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 258 - 258   2019.10

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  346. 遺伝性造血不全症候群に対する全エクソーム解析(Whole exome sequencing contributes to genetic diagnosis of IBMFS patients)

    Muramatsu Hideki, Hamada Motoharu, Okuno Yusuke, Wakamatsu Manabu, Taniguchi Rieko, Narita Kotaro, Miwata Shunsuke, Kitazawa Hironobu, Ichikawa Daisuke, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Ogi Tomoo, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 229 - 229   2019.10

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  347. 急性リンパ性白血病治療中に薬剤性神経障害により歩行困難となった14歳男児例(A case of a 14-year-old boy who had difficulty walking due to drug-induced neuropathy during T-cell acute lymphocytic leukemia treatment)

    Yoshida Taro, Sawamura Aya, Muramatsu Hideki, Imaya Masayuki, Yamamori Ayako, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Taniguchi Rieko, Ichikawa Daisuke, Nishikawa Eri, Hamada Motoharu, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Nakata Tomohiko, Kidokoro Hiroyuki, Natxume Jun, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 200 - 200   2019.10

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  348. 小児発熱性好中球減少症における次世代シークエンス病原微生物診断

    堀場 千尋, 鳥居 ゆか, 原 雄一郎, 嶋田 繭子, 鈴木 高子, 武内 俊, 奥村 俊彦, 川田 潤一, 村松 秀城, 高橋 義行, 荻 朋男, 伊藤 嘉規

    日本小児感染症学会総会・学術集会プログラム・抄録集   Vol. 51回   page: 196 - 196   2019.10

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  349. 再生不良性貧血における血漿中ウサギATG血中濃度と免疫抑制療法反応率の関連性(Correlation between plasma rabbit anti-thymocyte globulin level and response to immunosuppressive therapy in patients with severe aplastic anemia)

    Narita Atsushi, Muramatsu Hideki, Hamada Motoharu, Ichikawa Daisuke, Nishikawa Eri, Kawashima Nozomu, Okuno Yusuke, Nishio Nobuhiro, Yamazaki Hirohito, Nakao Shinji, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 213 - 213   2019.10

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  350. 初診時急性巨核芽球性白血病と鑑別を要した遺伝性葉酸吸収障害の乳児例(An infant with hereditary folate malabsorption requiring differentiation from acute megakaryoblastic leukemia)

    Nishikawa Eri, Taniguchi Rieko, Yoshida Taro, Imaya Masayuki, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Yamamori Ayako, Kitazawa Hironobu, Ichikawa Daisuke, Hamada Motoharu, Kawashima Nozomu, Narita Atsushi, Okuno Yusuke, Muramatsu Hideki, Nishio Nobuhiro, Kojima Seiji, Said Shimaa, Tozawa Yusuke, Yamada Masafumi, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 238 - 238   2019.10

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  351. 小児がん拠点病院におけるQuality Indicator

    藤崎 弘之, 小松 裕美, 井口 晶裕, 笹原 洋二, 康 勝好, 湯坐 有希, 後藤 裕明, 高橋 義行, 平山 雅浩, 滝田 順子, 家原 知子, 井上 雅美, 小阪 嘉之, 川口 浩史, 田口 智章, 木下 義晶, 米田 光宏, 瀧本 哲也, 松本 公一

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 343 - 343   2019.10

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  352. 小児悪性固形腫瘍における網羅的な融合遺伝子検出に基づいた遺伝子診断(Genetic diagnosis based on a comprehensive fusion gene analysis of pediatric malignant solid tumors)

    Ichikawa Daisuke, Muramatsu Hideki, Okuno Yusuke, Hamada Motoharu, Taniguchi Rieko, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Shimoyama Yoshie, Nakaguro Masato, Nakamura Shigeo, Kojima Seiji, Nakazawa Atsuko, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 227 - 227   2019.10

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  353. ホジキンリンパ腫治療後に治療関連急性前骨髄性白血病を発症しATRA+ATO療法で治療した1例

    山森 彩子, 成田 敦, 片岡 伸介, 濱田 太立, 市川 大輔, 川島 希, 西川 英里, 西尾 信博, 村松 秀城, 小島 勢二, 高橋 義行

    臨床血液   Vol. 60 ( 10 ) page: 1471 - 1471   2019.10

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  354. SIPOENスコアリングシステムを用いた、転移性高リスク神経芽腫に対するKIRリガンド不一致臍帯血移植の評価(Evaluation of the outcome of KIR-ligand mismatched cord blood transplantation for high risk metastatic neuroblastoma using new SIOPEN risk stratification)

    Nishio Nobuhiro, Ichikawa Daisuke, Wakamatsu Manabu, Narita Kotaro, Miwata Shunsuke, Kitazawa Hironobu, Taniguchi Rieko, Hamada Motoharu, Nishikawa Eri, Kawashima Nozomu, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 266 - 266   2019.10

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  355. がん免疫療法のUp to Date PiggyBacトランスポゾン法によるがんに対する遺伝子改変T細胞療法コスト削減の試み

    高橋 義行

    日本癌治療学会学術集会抄録集   Vol. 57回   page: SSY3 - 3   2019.10

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  356. ステロイド抵抗性腸管急性移植片対宿主病に対して経口ブデゾニド投与が有効であった2例(Two cases effectively treated with oral budesonide administration for steroid-resistant intestinal acute graft versus host disease)

    Yamamori Ayako, Nishikawa Eri, Narita Atsushi, Imaya Masayuki, Wakamatsu Manabu, Yoshida Taro, Miwata Shunsuke, Kitazawa Hironobu, Narita Kotaro, Taniguchi Rieko, Ichikawa Daisuke, Hamada Motoharu, Kawashima Nozomu, Okuno Yusuke, Nishio Nobuhiro, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 207 - 207   2019.10

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  357. セリチニブにより部分奏効が得られたALK転座陽性の治療抵抗性神経芽腫の1例(Induction of partial response with ceritinib for a child with chemotherapy-refractory ALK-rearranged neuroblastoma)

    Narita Kotaro, Yoshida Taro, Yamamori Ayako, Wakamatsu Manabu, Imaya Masayuki, Miwata Shunsuke, Kitazawa Hironobu, Taniguchi Rieko, Ichikawa Daisuke, Hamada Motoharu, Nishikawa Eri, Nishio Nobuhiro, Okuno Yusuke, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 264 - 264   2019.10

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  358. ファンコニ貧血診断における次世代シークエンス解析とFANCD2機能解析の統合の意義(Integrating FANCD2 functional analysis with high-throughput sequencing for diagnosing Fanconi anemia)

    Kawashima Nozomu, Okuno Yusuke, Muramatsu Hideki, Taniguchi Rieko, Yoshida Taro, Imaya Masayuki, Yamamori Ayako, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Hamada Motoharu, Ichikawa Daisuke, Kitazawa Hironobu, Nishikawa Eri, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 213 - 213   2019.10

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  359. KIRリガンド不一致臍帯血移植後神経芽腫中枢神経再発に対し、摘出及び全脳全脊髄照射により無増悪生存中の2例(Successful treatment of central nervous system relapses of neuroblastoma stage 4 after a KIR-ligand mismatched allogeneic cord blood transplantation)

    Imaya Masayuki, Narita Atsushi, Yoshida Taro, Yamamori Ayako, Wakamatsu Manabu, Miwata Syunsuke, Narita Kotaro, Kitazawa Hironobu, Taniguchi Rieko, Ichikawa Daisuke, Hamada Motoharu, Nishikawa Eri, Kawashima Nozomu, Muramatsu Hideki, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 263 - 263   2019.10

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  360. CAR-T細胞療法の基礎と臨床 急性リンパ性白血病に対する非ウイルスベクター法を用いたCD19標的CAR-T細胞療法の開発

    高橋 義行

    日本小児血液・がん学会雑誌   Vol. 56 ( 4 ) page: 165 - 165   2019.10

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  361. Delayed recognition of childhood arterial ischemic stroke. Reviewed International journal

    Ikumi Hori, Takeshi Tsuji, Misa Miyake, Kazuto Ueda, Erina Kataoka, Michio Suzuki, Satoru Kobayashi, Hirokazu Kurahashi, Yoshiyuki Takahashi, Akihisa Okumura, Tetsushi Yoshikawa, Shinji Saitoh, Jun Natsume

    Pediatrics international : official journal of the Japan Pediatric Society   Vol. 61 ( 9 ) page: 895 - 903   2019.9

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    BACKGROUND: Few population-based surveys of childhood arterial ischemic stroke (AIS) have been conducted in Asian countries. The aim of this study was to investigate the clinical features, time to diagnosis, and prognosis of childhood AIS in a population-based cohort in Japan. METHODS: Children aged 29 days-15 years 11 months old, residing in the Aichi Prefecture of Japan with radiologically confirmed AIS during 2010-2014, were identified retrospectively through questionnaires. We analyzed 40 children (23 boys, 17 girls; median age, 7 years 3 months), and collected time interval information of 26 patients. The time from clinical onset to first physician assessment and the time to AIS diagnosis were calculated. RESULTS: The most common presentation was paralysis or paresis in 27 patients (71%). No underlying disorders or possible trigger factors were identified in 14 patients (35%). The median time from symptom onset to first physician assessment was 2.9 h. The median time from symptom onset to the confirmed AIS diagnosis was 27.0 h. The diagnosis of AIS was made in the first 6 h after onset of symptoms in 27% of patients for whom the time was available. Radiological diagnosis took longer than 24 h in 54% of these patients. CONCLUSIONS: Long in-hospital delays exist in the diagnosis of AIS in children, likely due to lack of awareness of stroke by doctors. Further efforts to increase public and physician awareness of childhood stroke are needed to ensure early diagnosis and treatment.

    DOI: 10.1111/ped.13966

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  362. Risk factors for absence of catch-up growth in small for gestational age very low-birthweight infants. Reviewed International journal

    Sakiko Arai, Yoshiaki Sato, Hideki Muramatsu, Hidenori Yamamoto, Fumiko Aoki, Yu Okai, Shinsuke Kataoka, Yu Hanada, Motoharu Hamada, Yoshihito Morimoto, Seiji Kojima, Jun Natsume, Yoshiyuki Takahashi

    Pediatrics international : official journal of the Japan Pediatric Society   Vol. 61 ( 9 ) page: 889 - 894   2019.9

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    BACKGROUND: Many small for gestational age (SGA) infants have catch-up growth during the first 2 years of life, but approximately 10% have no catch-up growth, and short stature continues into adulthood. Identification of risk factors for absence of catch-up growth at an early age may be useful for earlier diagnosis and earlier treatment. METHODS: This was a retrospective multicenter study. The subjects were SGA infants with very low-birthweight (VLBW), who were followed up until the age of 3 years. The risk factors for absence of catch-up growth were identified on statistical analysis. RESULTS: Of the 217 SGA infants in this study, 181 were in the catch-up group and 36 were in the no catch-up group. The catch-up rate was 83%. On multivariate analysis adjusted for gestational age, birthweight, birth height, and birth head circumference, multipara, Z and ΔZ scores of length at 12 months of corrected age, and the Z score of height at 24 months of corrected age were risk factors for lack of catch-up at 3 years. CONCLUSIONS: The length Z and ΔZ scores at 12 months of corrected age may be useful for an earlier diagnosis and earlier initiation of growth hormone treatment in VLBW infants.

    DOI: 10.1111/ped.13939

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  363. 再発難治急性リンパ性白血病に対する新規治療戦略 次世代シークエンサーの臨床応用と遺伝子導入T細胞療法

    高橋 義行

    日本小児血液・がん学会雑誌   Vol. 56 ( 2 ) page: 244 - 244   2019.9

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  364. がんゲノム解析の新しい流れ(全ゲノム解析、クラウド解析、全がん解析、ロングリード解析などの新規技術) 若年性骨髄単球性白血病における全ゲノム解析(Frontiers in cancer genomics Whole-genome mutational analysis of juvenile myelomonocytic leukemia)

    奥野 友介, 村松 秀城, 村上 典寛, 川島 希, 若松 学, 北澤 宏展, 荻 朋男, 高橋 義行

    日本癌学会総会記事   Vol. 78回   page: S23 - 2   2019.9

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  365. SMARCA4不活化により発症し治療抵抗性を示した胸部腫瘍の1例

    市川 大輔, 村松 秀城, 奥野 友介, 若松 学, 三輪田 俊介, 成田 幸太郎, 北澤 宏展, 濱田 太立, 片岡 伸介, 村上 典寛, 谷口 理恵子, 鈴木 喬悟, 西川 英里, 西尾 信博, 川島 希, 成田 敦, 小島 勢二, 高橋 義行, 中黒 匡人, 中村 栄男, 中澤 温子

    日本小児血液・がん学会雑誌   Vol. 56 ( 2 ) page: 246 - 246   2019.9

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  366. CAR-T細胞の臨床応用 急性リンパ性白血病に対する非ウイルスベクター法を用いたCAR-T細胞療法の開発(CAR-T in Clinics Development of CAR-T cell therapy for acute lymphoblastic leukemia using non-virus vector system)

    高橋 義行

    日本癌学会総会記事   Vol. 78回   page: S5 - 2   2019.9

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  367. Constitutive activation of mTORC1 signaling induced by biallelic loss-of-function mutations in SZT2 underlies a discernible neurodevelopmental disease. Reviewed International journal

    Yuji Nakamura, Kohji Kato, Naomi Tsuchida, Naomichi Matsumoto, Yoshiyuki Takahashi, Shinji Saitoh

    PloS one   Vol. 14 ( 8 ) page: e0221482   2019.8

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    There have been increasing number of reports of SZT2-related neurological diseases, the main symptoms of which are epilepsy, developmental delay, macrocephaly and a dysmorphic corpus callosum. SZT2 functions as a regulator of mechanistic target of rapamycin complex 1 (mTORC1) signaling in cultured human cell lines and mouse tissues. However, it remains to be determined whether mutations in SZT2 in human patients alter mTORC1 signaling. In this study, we aimed to investigate the functional consequence of biallelic SZT2 variants in Epstein-Barr virus-induced lymphoblastoid cell lines (LCLs) established from two patients with a typical SZT2-related neurodevelopmental disease. Increased phosphorylation of S6 kinase and S6 was identified in patient-derived cell lines under amino acid-starved condition, suggestive of constitutive hyperactivation of mTORC1 signaling. This result was validated by constitutive lysosomal localization of mTOR in patients' LCLs. Furthermore, patients' LCLs display an excessive response to slight amino acid stimulation. Our data suggest the loss-of-function nature of SZT2 mutations in the patients, and consequent hyperactivation of mTORC1 signaling in response to both amino acid starvation and stimulation in their LCLs. By these functional analyses, the pathogenicity of newly identified SZT2 variants can be determined, allowing for more detailed characterization of genotype-phenotype correlations.

    DOI: 10.1371/journal.pone.0221482

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  368. Impact of low-dose irradiation and in vivo T-cell depletion on hematopoietic stem cell transplantation for non-malignant diseases using fludarabine-based reduced-intensity conditioning. Reviewed International journal

    Katsutsugu Umeda, Hiromasa Yabe, Koji Kato, Kohsuke Imai, Masao Kobayashi, Yoshiyuki Takahashi, Nao Yoshida, Maho Sato, Yoji Sasahara, Keisuke Kato, Souichi Adachi, Yuhki Koga, Keiko Okada, Masami Inoue, Yoshiko Hashii, Yoshiko Atsuta, Tomohiro Morio

    Bone marrow transplantation   Vol. 54 ( 8 ) page: 1227 - 1236   2019.8

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    Reduced-intensity conditioning is widely used with hematopoietic stem cell transplantation for non-malignant diseases: however, the optimal conditioning to ensure stable engraftment has not been established. In this study, we retrospectively compared the impact of low-dose (1-6 Gy) irradiation and in vivo T-cell depletion on the clinical outcome of 523 patients with non-malignant disease who underwent a first allogeneic hematopoietic stem cell transplantation using fludarabine-based reduced-intensity conditioning. Use of low-dose irradiation, but not of anti-thymocyte globulin/anti-lymphocyte globulin, showed a beneficial effect on overall survival (adjusted hazard ratio: 0.56; 95% confidence interval: 0.35-0.91, P = 0.018). Furthermore, use of low-dose irradiation was strongly associated with lower transplant-related mortality (adjusted hazard ratio: 0.55; 95% confidence interval: 0.32-0.96, P = 0.034). The addition of low-dose irradiation to the conditioning regimen was beneficial, at least to the short-term clinical outcome. A large prospective study with long-term follow-up is now required to extend these findings and establish the optimal hematopoietic stem cell transplant conditioning for patients with at least some subgroups of non-malignant diseases.

    DOI: 10.1038/s41409-018-0418-8

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  369. 愛知県におけるチャイルド・デス・レビューの試み

    沼口 敦, 高橋 義行, 夏目 淳, 伊藤 嘉規, 齋藤 伸治, 吉川 哲史, 奥村 彰久

    日本小児科学会雑誌   Vol. 123 ( 8 ) page: 1313 - 1313   2019.8

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  370. 小児重症呼吸不全患者の気管支肺胞洗浄液を用いた次世代シークエンス法による病原微生物の網羅的検出

    武内 俊, 川田 潤一, 堀場 千尋, 奥村 俊彦, 鈴木 高子, 鳥居 ゆか, 奥野 友介, 高橋 義行, 伊藤 嘉規, 河邉 慎司, 池山 貴也

    日本小児科学会雑誌   Vol. 123 ( 8 ) page: 1336 - 1336   2019.8

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  371. 先天性嚢胞性肺疾患との鑑別に苦慮した巨大縦隔気腫の2例

    本部 和也, 脇田 浩正, 上田 一仁, 田中 亮, 三浦 良介, 呉 尚治, 浅田 英之, 北瀬 悠磨, 鈴木 俊彦, 兵藤 玲奈, 見松 はるか, 伊藤 美春, 齊藤 明子, 村松 友佳子, 佐藤 義朗, 早川 昌弘, 高橋 義行

    日本小児科学会雑誌   Vol. 123 ( 8 ) page: 1312 - 1312   2019.8

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  372. ステロイド減量に伴い再発を繰り返した抗MOG抗体陽性急性散在性脳脊髄炎(ADEM)の1例

    隈井 すみれ, 山本 啓之, 川口 将宏, 牧 祐輝, 田中 雅大, 岡井 佑, 坂口 陽子, 中田 智彦, 城所 博之, 高橋 義行, 夏目 淳, 濱口 直子, 柴田 元博, 金子 仁彦, 高橋 利幸

    日本小児科学会雑誌   Vol. 123 ( 8 ) page: 1310 - 1310   2019.8

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  373. テクノロジーの進展は検査医学をどう変えるか 急性リンパ性白血病に対する次世代シークエンサーを用いた遺伝子診断およびキメラ抗原受容体遺伝子改変T(CAR-T)細胞療法

    高橋 義行

    臨床病理   Vol. 67 ( 8 ) page: 884 - 884   2019.8

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  374. CF290 for pancolonic chromoendoscopy improved sessile serrated polyp detection and procedure time: a propensity score-matching study. Reviewed

    Toyoshima O, Yoshida S, Nishizawa T, Yamakawa T, Sakitani K, Hata K, Takahashi Y, Fujishiro M, Watanabe H, Koike K

    Endoscopy international open   Vol. 7 ( 8 ) page: E987 - E993   2019.8

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    DOI: 10.1055/a-0953-1909

    PubMed

  375. Whole abdominopelvic intensity-modulated radiation therapy for peritoneal disseminated rhabdomyosarcoma with three-year follow-up: a case report. Reviewed International journal

    Mariko Kawamura, Kuniyasu Okudaira, Yoshiyuki Itoh, Takeshi Kamomae, Eri Nishikawa, Hideki Muramatsu, Yoshiyuki Takahashi, Kazuki Yokota, Shinji Naganawa

    Radiation oncology (London, England)   Vol. 14 ( 1 ) page: 127 - 127   2019.7

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    BACKGROUND: The role of local radiotherapy in the treatment of metastatic rhabdomyosarcoma is important. However, with peritoneal dissemination, the application of local therapy is challenging. Although there are few reports addressing the efficacy of the whole abdominopelvic irradiation to peritoneal disseminated rhabdomyosarcoma patients, no precise curse of treatment nor the follow up result is explained in paper nor in the text. CASE PRESENTATION: Six years old rhabdomyosarcoma boy with peritoneal dissemination was treated at our facility under COG D9803 protocol (vincristine, dactinomycin, and cyclophosphamide (VAC)). He underwent tumor resection on the 14th week according to the protocol. During surgery, the 2-cm residual tumor was completely resected, but in the pelvis, numerous nodules that were suspected as peritoneal disseminated tumors were observed. We administered 30 Gy/20fr whole abdominopelvic radiotherapy using volumetric modulated arc therapy (VMAT) technique and a 6 Gy sequential boost to pelvis after the surgery and completed the protocol treatment. During the course of treatment, the patient experienced G4 hematological toxicity and received multiple transfusions, particularly after whole abdominopelvic irradiation. He has achieved complete remission and is alive without evidence of recurrence and severe late adverse effect for 3 years. In terms of growth, his height and weight are within the average values for Japanese boys at the same age. CONCLUSION: By using the VMAT technique, a patient with peritoneal disseminated rhabdomyosarcoma can be treated, and a dose of 30 Gy to the whole abdominopelvis with concurrent chemotherapy may be tolerable.

    DOI: 10.1186/s13014-019-1333-x

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  376. Comparison of Clinical Characteristics of Human Metapneumovirus and Respiratory Syncytial Virus Infections in Hospitalized Young Children. Reviewed

    Akinobu Taniguchi, Jun-Ichi Kawada, Kiyotaka Go, Naozumi Fujishiro, Yosuke Hosokawa, Yuki Maki, Yuichiro Sugiyama, Michio Suzuki, Takeshi Tsuji, Shin Hoshino, Hideki Muramatsu, Hiroyuki Kidokoro, Fumie Kinoshita, Akihiro Hirakawa, Yoshiyuki Takahashi, Yoshiaki Sato, Jun Natsume

    Japanese journal of infectious diseases   Vol. 72 ( 4 ) page: 237 - 242   2019.7

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    Human metapneumovirus (HMPV) and respiratory syncytial virus (RSV) are the leading causes of acute respiratory tract infection in children, and clinical manifestations of these virus infections are considered similar. To investigate the differences in clinical characteristics between HMPV and RSV infections in young children, we prospectively enrolled children < 3 years old who required hospitalization with acute respiratory tract infection due to HMPV or RSV at 10 hospitals in Japan. We enrolled 48 children with HMPV infection and 141 with RSV infection. Patients with HMPV infection were older than those with RSV infection. High-grade fever was more frequently observed in patients with HMPV infection, whereas no significant differences in respiratory symptoms were apparent. Abnormal serum lactate dehydrogenase values and consolidation shadows on chest X-ray were more frequently observed in patients with HMPV infection. During hospitalization, nasal mucus suction was more frequently required in patients with RSV infection. On the other hand, β2-adrenergic agonists, corticosteroids, and leukotriene receptor antagonists were more frequently used in patients with HMPV infection. These findings suggest that HMPV and RSV infections show similar respiratory symptoms, but HMPV infection is more likely to lead to the development of pneumonia, at least among hospitalized young children.

    DOI: 10.7883/yoken.JJID.2018.480

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  377. Effects of HLA mismatch on cytomegalovirus reactivation in cord blood transplantation

    Yokoyama, H; Kanda, J; Kato, S; Kondo, E; Maeda, Y; Saji, H; Takahashi, S; Onizuka, M; Onishi, Y; Ozawa, Y; Kanamori, H; Ishikawa, J; Ohno, Y; Ichinohe, T; Takanashi, M; Kato, K; Atsuta, Y; Kanda, Y; Ikegame, K; Utsunomiya, A; Kawase, T; Kim, SW; Kuwatsuka, Y; Kobayashi, T; Takatsuka, Y; Takahashi, Y; Tanaka, J; Tamaki, H; Tsuji, M; Nishida, T; Masuko, M; Matsuno, R; Murata, M; Morishima, S; Morishima, Y; Yokoyama, H; Wake, A; Watanabe, N; Ashida, T; Hoshino, T; Yabe, T; Sakamoto, K; Fuji, S; Miyamura, K; Arima, N; Kondo, E; Yoshimitsu, M; Kawamura, K; Kawata, T; Kishimoto, K; Tatara, R; Hagino, T; Fujiwara, SI; Shimomura, Y; Sakaguchi, H; Hirabayashi, S; Ishii, H; Onda, Y; Kato, I; Kawajiri, A; Shindo, T; Tokunaga, M; Nonami, A; Muranushi, H; Yoshinaga, N; Kawashima, N; Shiratori, S; Tada, Y; Tanoue, SU; Hirayama, M; Fukunaga, K; Ohbiki, M

    BONE MARROW TRANSPLANTATION   Vol. 54 ( 7 ) page: 1004 - 1012   2019.7

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    DOI: 10.1038/s41409-018-0369-0

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  378. Relationship between plasma rabbit anti-thymocyte globulin level and response to immunosuppressive therapy in patients with severe aplastic anemia: Results of a multicenter, prospective, randomized study

    Narita, A; Muramatsu, H; Xu, YY; Nishikawa, E; Kawashima, N; Okuno, Y; Nishio, N; Hama, A; Yamazaki, H; Nakao, S; Kojima, S; Takahashi, Y

    BONE MARROW TRANSPLANTATION   Vol. 54   page: 29 - 29   2019.7

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  379. Relationship between plasma rabbit anti-thymocyte globulin level and response to immunosuppressive therapy in patients with severe aplastic anemia: Results of a multicenter, prospective, randomized study

    Narita Atsushi, Muramatsu Hideki, Xu Yinyan, Nishikawa Eri, Kawashima Nozomu, Okuno Yusuke, Nishio Nobuhiro, Hama Asahito, Yamazaki Hirohito, Nakao Shinji, Kojima Seiji, Takahashi Yoshiyuki

    BONE MARROW TRANSPLANTATION   Vol. 54   page: 29-29   2019.7

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  380. MYCN de novo gain-of-function mutation in a patient with a novel megalencephaly syndrome. Reviewed International journal

    Kohji Kato, Fuyuki Miya, Nanako Hamada, Yutaka Negishi, Yoko Narumi-Kishimoto, Hiroshi Ozawa, Hidenori Ito, Ikumi Hori, Ayako Hattori, Nobuhiko Okamoto, Mitsuhiro Kato, Tatsuhiko Tsunoda, Yonehiro Kanemura, Kenjiro Kosaki, Yoshiyuki Takahashi, Koh-Ichi Nagata, Shinji Saitoh

    Journal of medical genetics   Vol. 56 ( 6 ) page: 388 - 395   2019.6

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    BACKGROUND: In this study, we aimed to identify the gene abnormality responsible for pathogenicity in an individual with an undiagnosed neurodevelopmental disorder with megalencephaly, ventriculomegaly, hypoplastic corpus callosum, intellectual disability, polydactyly and neuroblastoma. We then explored the underlying molecular mechanism. METHODS: Trio-based, whole-exome sequencing was performed to identify disease-causing gene mutation. Biochemical and cell biological analyses were carried out to elucidate the pathophysiological significance of the identified gene mutation. RESULTS: We identified a heterozygous missense mutation (c.173C>T; p.Thr58Met) in the MYCN gene, at the Thr58 phosphorylation site essential for ubiquitination and subsequent MYCN degradation. The mutant MYCN (MYCN-T58M) was non-phosphorylatable at Thr58 and subsequently accumulated in cells and appeared to induce CCND1 and CCND2 expression in neuronal progenitor and stem cells in vitro. Overexpression of Mycn mimicking the p.Thr58Met mutation also promoted neuronal cell proliferation, and affected neuronal cell migration during corticogenesis in mouse embryos. CONCLUSIONS: We identified a de novo c.173C>T mutation in MYCN which leads to stabilisation and accumulation of the MYCN protein, leading to prolonged CCND1 and CCND2 expression. This may promote neurogenesis in the developing cerebral cortex, leading to megalencephaly. While loss-of-function mutations in MYCN are known to cause Feingold syndrome, this is the first report of a germline gain-of-function mutation in MYCN identified in a patient with a novel megalencephaly syndrome similar to, but distinct from, CCND2-related megalencephaly-polymicrogyria-polydactyly-hydrocephalus syndrome. The data obtained here provide new insight into the critical role of MYCN in brain development, as well as the consequences of MYCN defects.

    DOI: 10.1136/jmedgenet-2018-105487

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  381. 愛知県におけるチャイルド・デス・レビュー 調査から検証へ

    沼口 敦, 夏目 淳, 齋藤 伸治, 吉川 哲史, 奥村 彰久, 高橋 義行

    日本小児救急医学会雑誌   Vol. 18 ( 2 ) page: 262 - 262   2019.6

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  382. 厳しい予後の告知を受け、子ども自身が終末期の意思決定を行った高校生の一例

    佐々木 美和, 滝 良梨子, 梶山 早苗, 野田 弘実, 谷口 理恵子, 上田 一仁, 村松 秀城, 高橋 義行

    Palliative Care Research   Vol. 14 ( Suppl. ) page: S346 - S346   2019.6

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  383. Comparison of high-dose and low-dose corticosteroid therapy for refractory Mycoplasma pneumoniae pneumonia in children. Reviewed International journal

    Toshihiko Okumura, Jun-Ichi Kawada, Masaharu Tanaka, Kotaro Narita, Tomonori Ishiguro, Yuji Hirayama, Sho Narahara, Genki Tsuji, Yuichiro Sugiyama, Michio Suzuki, Takeshi Tsuji, Shin Hoshino, Masahiro Nakatochi, Hideki Muramatsu, Hiroyuki Kidokoro, Yoshiyuki Takahashi, Yoshiaki Sato

    Journal of infection and chemotherapy : official journal of the Japan Society of Chemotherapy   Vol. 25 ( 5 ) page: 346 - 350   2019.5

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    BACKGROUND: Mycoplasma pneumoniae pneumonia (MPP) is generally a self-limiting disease, but it may become refractory. It is thought that refractory MPP is linked to the excessive immunologic responses of the host. Consequently, the use of adjunctive systemic corticosteroids may have beneficial effects. In this study, we compared the effects of high- and low-dose corticosteroid therapy in a pediatric population with refractory MPP. METHODS: We retrospectively collected data from 91 pediatric MPP patients treated with adjunctive systemic corticosteroids between April 2014 and October 2016. The patients were divided into the following two groups: high-dose corticosteroid group (2 mg/kg/day or more of prednisolone equivalents; n = 38) and low-dose corticosteroid group (<2 mg/kg/day; n = 53). Additionally, we compared the number of febrile days post-corticosteroid administration. We used 25 paired patients in a propensity score matching analysis to correct for confounding factors both by age and by days (from onset till corticosteroid therapy initiation). RESULTS: We observed that in the high-dose corticosteroid group defervescence following corticosteroid therapy initiation was achieved significantly earlier and length of hospitalization was significantly shorter (0.8 ± 1.0 vs. 1.5 ± 1.4 days and 8.2 ± 2.4 vs. 10.7 ± 2.7 days, respectively). In the propensity score matching, we observed that significant differences in the length of fever following corticosteroid therapy initiation and hospitalization were still present. Further, neither of the groups developed corticosteroid-related adverse events. CONCLUSION: Our results suggest that patients with refractory MPP treated with high-dose corticosteroid could achieve defervescence earlier and have a shorter hospitalization.

    DOI: 10.1016/j.jiac.2019.01.003

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  384. High rates of ovarian function preservation after hematopoietic cell transplantation with melphalan-based reduced intensity conditioning for pediatric acute leukemia: an analysis from the Japan Association of Childhood Leukemia Study (JACLS). Reviewed

    Hisanori Fujino, Hiroyuki Ishida, Akihiro Iguchi, Masaei Onuma, Koji Kato, Mariko Shimizu, Masahiro Yasui, Hiroyuki Fujisaki, Kazuko Hamamoto, Kana Washio, Hirotoshi Sakaguchi, Emiko Miyashita, Yuko Osugi, Etsuko Nakagami-Yamaguchi, Akira Hayakawa, Atsushi Sato, Yoshiyuki Takahashi, Keizo Horibe

    International journal of hematology   Vol. 109 ( 5 ) page: 578 - 583   2019.5

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    Women are at high risk of hypergonadotropic hypogonadism after hematopoietic cell transplantation (HCT). Hypogonadism is universal after irradiation or busulfan. We hypothesized that reduced intensity conditioning (RIC) might protect ovarian function after HCT. We retrospectively reviewed data from patients with acute leukemia treated according to the Japan Association of Childhood Leukemia Study and nationwide multicenter study protocol. We selected 11 female patients with acute leukemia who received first HCT with RIC, had survived for three or more years after HCT, and were aged ≥ 12 years at the last follow-up visit. Median age at diagnosis, HCT, and last visit were 8, 10, and 17 years. Six patients received HLA-matched bone marrow (BM), two HLA-mismatched BM, and three cord blood. Melphalan was used as conditioning regimen in all patients. At the last visit, six of seven post-pubertal patients at transplantation recovered menstruation, and four of four patients who underwent transplantation at the pre-pubertal began menstruation. Height z scores showed no significant reduction between pre-transplant and post-transplant. No patients received growth hormone treatment. Only one recipient displayed subclinical hypothyroidism. Melphalan-based RIC may be an encouraging option for patients with acute leukemia to avoid ovarian and endocrine dysfunction after HCT.

    DOI: 10.1007/s12185-019-02627-9

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  385. Design of a prospective multicenter randomized controlled trial evaluating the effects of gastric lavage on coffee-ground emesis in neonates: study protocol. Reviewed

    Takashi Maeda, Yoshiaki Sato, Akihiro Hirakawa, Masahiro Nakatochi, Fumie Kinoshita, Takeshi Suzuki, Shintaro Ichimura, Ryoichi Ito, Ryuji Kudo, Michio Suzuki, Shin Hoshino, Yuichiro Sugiyama, Hideki Muramatsu, Hiroyuki Kidokoro, Jun-Ichi Kawada, Yoshiyuki Takahashi

    Nagoya journal of medical science   Vol. 81 ( 2 ) page: 227 - 232   2019.5

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    Neonates who swallow a considerable amount of maternal blood may exhibit vomiting and suckling disorder during the first few days of the postnatal period. Some clinicians treat these neonates with gastric lavage (GL) to prevent vomiting and the establishment of enteral feeding empirically, but there was no study assessing the effect of GL for neonates with coffee-ground emesis. We designed a multicenter randomized controlled trial to evaluate the efficacy and safety of GL in neonates with coffee-ground emesis. Vigorous neonates with birth weight ranging from 2500 g to 3999 g and gestational age between 37w0d and 41w6d who presented with coffee-ground emesis on more than twice and diagnosed as false melena, were divided into two groups using computerized randomization. We defined feeding intolerance (FI) as (1) ≥2 vomiting episodes in 4h or ≥3 episodes in 24h and/or (2) feeding failure on at least two occasions because of retching or poor sucking. Primary outcome is percentage of infants who present FI within 24 hours from admission. We also assessed the residual volumes, number of vomiting episodes, percentage of weight reduction at postnatal day 4, rates of body weight gain at 1 month of age, and peak serum total bilirubin value before discharge. To our knowledge, this is the first study to evaluate the safety and efficacy of GL for neonates with coffee-ground emesis. This trial is registered at UMIN Clinical Trials Registry as UMIN000026483.

    DOI: 10.18999/nagjms.81.2.227

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  386. Successful hematopoietic stem cell transplantation from an HLA-mismatched parent for engraftment failure after unrelated cord blood transplantation in patients with juvenile myelomonocytic leukemia: Report of two cases. Reviewed International journal

    Koshi Akahane, Atsushi Watanabe, Yoshiyuki Furuichi, Shinpei Somazu, Hiroko Oshiro, Kumiko Goi, Kazuo Sakashita, Hideki Muramatsu, Asahito Hama, Yoshiyuki Takahashi, Kenichi Koike, Seiji Kojima, Kanji Sugita, Takeshi Inukai

    Pediatric transplantation   Vol. 23 ( 3 ) page: e13378   2019.5

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    JMML is an aggressive hematopoietic malignancy of early childhood, and allogeneic HSCT is the only curative treatment for this disease. Umbilical cord blood is one of donor sources for HSCT in JMML patients who do not have an HLA-compatible relative, but engraftment failure remains a major problem. Here, we report two cases of JMML who were successfully rescued by HSCT from an HLA-mismatched parent after development of primary engraftment failure following unrelated CBT. Both patients had severe splenomegaly and underwent unrelated CBT from an HLA-mismatched donor. Immediately after diagnosis of engraftment failure, both patients underwent HSCT from their parent. For the second HSCT, we used RIC regimens consisting of FLU, CY, and a low dose of rabbit ATG with or without TBI and additionally administered ETP considering their persistent severe splenomegaly. Both patients achieved engraftment without severe treatment-related adverse effects. After engraftment of second HSCT, their splenomegaly was rapidly regressed, and both patients showed no sign of relapse for over 4 years. These observations demonstrate that HSCT from an HLA-mismatched parent could be a feasible salvage treatment for primary engraftment failure in JMML patients.

    DOI: 10.1111/petr.13378

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  387. 難治性マイコプラズマ肺炎に対するステロイド療法の検討

    奥村 俊彦, 川田 潤一, 田中 雅大, 成田 幸太郎, 石黒 智紀, 平山 祐司, 楢原 翔, 辻 元基, 松村 秀城, 城所 博之, 佐藤 義朗, 高橋 義行, 杉山 裕一朗, 鈴木 道雄, 辻 健史, 星野 伸

    日本小児科学会雑誌   Vol. 123 ( 5 ) page: 912 - 913   2019.5

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  388. 左頸部の軟属腫様皮疹から診断した多臓器型Langerhans cell histiocytosis(LCH)の1例

    太田 早紀, 桃原 真理子, 河野 通浩, 秋山 真志, 北澤 宏展, 高橋 義行

    日本皮膚科学会雑誌   Vol. 129 ( 6 ) page: 1360 - 1360   2019.5

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  389. 小児血液・腫瘍医からみた血液・がん疾患に関連する皮膚病変

    川島 希, 高橋 義行

    日本皮膚科学会雑誌   Vol. 129 ( 5 ) page: 1069 - 1069   2019.5

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  390. 非ウイルスベクター法を用いたキメラ抗原受容体遺伝子導入T細胞(CAR-T細胞)療法の現状と展望

    高橋 義行

    日本医学会総会会誌   Vol. 30回   page: LS - 1   2019.4

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  391. 可能な限り鎮静を減らすために 4歳児が鎮静なく放射線治療ができた関わり

    村嶋 一歩, 佐々木 美和, 野田 弘実, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 56 ( 1 ) page: 74 - 74   2019.4

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  392. 友達の死を他の患児へ伝え、グリーフケアにつなげる取り組み

    牧田 夏美, 佐々木 美和, 萩原 沙織, 和田 拓哉, 森本 綾, 西川 英里, 高橋 義行, 柴田 有理, 河野 智子

    日本小児血液・がん学会雑誌   Vol. 56 ( 1 ) page: 74 - 75   2019.4

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  393. Discontinuation of L-asparaginase and poor response to prednisolone are associated with poor outcome of ETV6-RUNX1-positive pediatric B-cell precursor acute lymphoblastic leukemia. Reviewed

    Usami I, Imamura T, Takahashi Y, Suenobu SI, Hasegawa D, Hashii Y, Deguchi T, Hori T, Shimada A, Kato K, Ito E, Moriya-Saito A, Kawasaki H, Hori H, Yumura-Yagi K, Hara J, Sato A, Horibe K, Japan, Association of Childhood Leukemia Study Group (JACLS

    International journal of hematology   Vol. 109 ( 4 ) page: 477 - 482   2019.4

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    ETV6-RUNX1-positive B precursor acute lymphoblastic leukemia (B-ALL) is a common subtype of pediatric B-ALL that has shown excellent outcomes in contemporary clinical trials for pediatric B-ALL. Examinations of the possibility of reducing therapeutic intensity may thus be explored. This prospective study examined outcomes in 205 pediatric patients with ETV6-RUNX1-positive B-ALL uniformly treated following the Japan Association of Childhood Leukemia Study Group (JACLS) ALL-02 protocol. The JACLS ALL-02 protocol does not employ minimal residual disease detected by polymerase chain reaction (PCR-MRD)-based risk stratification; however, 4-year event-free survival (EFS) and overall survival (OS) were 94.4 ± 1.6 and 97.5 ± 1.1%, respectively. In particular, 92 of 205 (44.9%) patients were successfully treated with a less intensive regimen involving only two cycles of high dose methotrexate and one course of re-induction therapy comprising vincristine, L-asparaginase (L-asp), pirarubicin, and prednisolone. Multivariate analysis revealed that discontinuation of L-asp and poor response to prednisolone was, respectively, associated with poor EFS (HR 6.3; 95% CI 1.3-27.0) and OS (HR 17.5; 95% CI 2.3-130), suggesting that the majority of ETV6-RUNX1-positive B-ALL cases may be cured by a less-intensive chemotherapy regimen if the risk stratification system including PCR-MRD monitoring and insufficient use of L-asp is avoided.

    DOI: 10.1007/s12185-019-02599-w

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  394. A combination of serum anti-<i>Helicobacter pylori</i> antibody titer and Kyoto classification score could provide a more accurate diagnosis of <i>H pylori</i>. Reviewed International journal

    Nishizawa T, Sakitani K, Suzuki H, Yamakawa T, Takahashi Y, Yamamichi N, Watanabe H, Seto Y, Koike K, Toyoshima O

    United European gastroenterology journal   Vol. 7 ( 3 ) page: 343 - 348   2019.4

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    BACKGROUND: We previously showed that the endoscopic Kyoto classification for gastritis could predict Helicobacter pylori infection in individuals with a high negative titer of serum anti-H pylori antibodies. This study evaluated H pylori infection and the Kyoto classification score in patients with a low negative titer (<3 U/ml), high negative titer (3-9.9 U/ml), low positive titer (10-49.9 U/ml), and high positive titer (≥50 U/ml). METHODS: Serum antibody levels, Kyoto classification score and histology were investigated in 870 individuals with no history of H pylori-eradication therapy. Urea breath tests (UBTs) were additionally conducted for patients with a low negative titer and a Kyoto score ≥1 or an antibody titer ≥10 U/ml and a Kyoto score of 0 or 1. UBTs and/or histological studies were conducted for participants with a high negative titer. RESULTS: False diagnoses based on anti-H pylori antibody titers were observed in 0.3% of the low-negative-titer group, 11.7% of the high-negative-titer group, 18.9% of the low-positive-titer group and 2.2% of the high-positive-titer group. Surprisingly, false diagnoses based on antibody titers were noted in 63.2% of patients with a low positive titer and a Kyoto score of 0 and in 62.5% of patients with a high negative titer and a Kyoto score ≥2, respectively. CONCLUSIONS: Endoscopic findings could predict false diagnoses determined using serum antibody titers.

    DOI: 10.1177/2050640619825947

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  395. AYA世代を迎えた小児血液疾患・腫瘍疾患経験者の患者会の発足と活動報告

    太田 晃嗣, 佐々木 美和, 清水 直子, 秋田 麻衣, 谷口 理恵子, 西川 英里, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 56 ( 1 ) page: 73 - 73   2019.4

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  396. Defective Epstein-Barr virus in chronic active infection and haematological malignancy. Reviewed International journal

    Yusuke Okuno, Takayuki Murata, Yoshitaka Sato, Hideki Muramatsu, Yoshinori Ito, Takahiro Watanabe, Tatsuya Okuno, Norihiro Murakami, Kenichi Yoshida, Akihisa Sawada, Masami Inoue, Keisei Kawa, Masao Seto, Koichi Ohshima, Yuichi Shiraishi, Kenichi Chiba, Hiroko Tanaka, Satoru Miyano, Yohei Narita, Masahiro Yoshida, Fumi Goshima, Jun-Ichi Kawada, Tetsuya Nishida, Hitoshi Kiyoi, Seiichi Kato, Shigeo Nakamura, Satoko Morishima, Tetsushi Yoshikawa, Shigeyoshi Fujiwara, Norio Shimizu, Yasushi Isobe, Masaaki Noguchi, Atsushi Kikuta, Keiji Iwatsuki, Yoshiyuki Takahashi, Seiji Kojima, Seishi Ogawa, Hiroshi Kimura

    Nature microbiology   Vol. 4 ( 3 ) page: 404 - 413   2019.3

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    Epstein-Barr virus (EBV) infection is highly prevalent in humans and is implicated in various diseases, including cancer1,2. Chronic active EBV infection (CAEBV) is an intractable disease classified as a lymphoproliferative disorder in the 2016 World Health Organization lymphoma classification1,2. CAEBV is characterized by EBV-infected T/natural killer (NK) cells and recurrent/persistent infectious mononucleosis-like symptoms3. Here, we show that CAEBV originates from an EBV-infected lymphoid progenitor that acquires DDX3X and other mutations, causing clonal evolution comprising multiple cell lineages. Conspicuously, the EBV genome in CAEBV patients harboured frequent intragenic deletions (27/77) that were also common in various EBV-associated neoplastic disorders (28/61), including extranodal NK/T-cell lymphoma and EBV-positive diffuse large B-cell lymphoma, but were not detected in infectious mononucleosis or post-transplant lymphoproliferative disorders (0/47), which suggests a unique role of these mutations in neoplastic proliferation of EBV-infected cells. These deletions frequently affected BamHI A rightward transcript microRNA clusters (31 cases) and several genes that are essential for producing viral particles (20 cases). The deletions observed in our study are thought to reactivate the lytic cycle by upregulating the expression of two immediate early genes, BZLF1 and BRLF14-7, while averting viral production and subsequent cell lysis. In fact, the deletion of one of the essential genes, BALF5, resulted in upregulation of the lytic cycle and the promotion of lymphomagenesis in a xenograft model. Our findings highlight a pathogenic link between intragenic EBV deletions and EBV-associated neoplastic proliferations.

    DOI: 10.1038/s41564-018-0334-0

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  397. Publisher Correction: Defective Epstein-Barr virus in chronic active infection and haematological malignancy. Reviewed International journal

    Yusuke Okuno, Takayuki Murata, Yoshitaka Sato, Hideki Muramatsu, Yoshinori Ito, Takahiro Watanabe, Tatsuya Okuno, Norihiro Murakami, Kenichi Yoshida, Akihisa Sawada, Masami Inoue, Keisei Kawa, Masao Seto, Koichi Ohshima, Yuichi Shiraishi, Kenichi Chiba, Hiroko Tanaka, Satoru Miyano, Yohei Narita, Masahiro Yoshida, Fumi Goshima, Jun-Ichi Kawada, Tetsuya Nishida, Hitoshi Kiyoi, Seiichi Kato, Shigeo Nakamura, Satoko Morishima, Tetsushi Yoshikawa, Shigeyoshi Fujiwara, Norio Shimizu, Yasushi Isobe, Masaaki Noguchi, Atsushi Kikuta, Keiji Iwatsuki, Yoshiyuki Takahashi, Seiji Kojima, Seishi Ogawa, Hiroshi Kimura

    Nature microbiology   Vol. 4 ( 3 ) page: 544 - 544   2019.3

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    In the version of this Letter originally published, in the sentence beginning "The major driver role of DDX3X mutations...", the citation "Fig. 2a-f" should have been "Fig. 2". In addition, in the sentence beginning "Another finding of interest was the presence of identical driver mutations...", the citation "Fig. 3a,b and Fig. 4" should have been "Fig. 3". This has now been amended in all versions of the Letter.

    DOI: 10.1038/s41564-019-0387-8

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  398. 次世代シークエンス解析による発熱性好中球減少症の網羅的病原微生物検出

    堀場 千尋, 鳥居 ゆか, 鈴木 高子, 武内 俊, 奥村 俊彦, 川田 潤一, 伊藤 嘉規, 高橋 義行, 嶋田 繭子, 荻 朋男

    感染症学雑誌   Vol. 93 ( 臨増 ) page: 325 - 325   2019.3

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  399. がん診療拠点病院における整形外科 -その現状と課題- 小児がん拠点病院における整形外科診療 小児がん治療後の整形外科的問題点と小児期の骨・軟部肉腫

    西田 佳弘, 内田 広夫, 高橋 義行, 石黒 直樹

    日本整形外科学会雑誌   Vol. 93 ( 2 ) page: S371 - S371   2019.3

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  400. Frequent intragenic microdeletions of elastin in familial supravalvular aortic stenosis. Reviewed International journal

    Satoshi Hayano, Yusuke Okuno, Makiko Tsutsumi, Hidehito Inagaki, Yoshie Fukasawa, Hiroki Kurahashi, Seiji Kojima, Yoshiyuki Takahashi, Taichi Kato

    International journal of cardiology   Vol. 274   page: 290 - 295   2019.1

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    BACKGROUND: Supravalvular aortic stenosis (SVAS) is a congenital heart disease affecting approximately 1:25,000 live births. SVAS may occur sporadically, be inherited in an autosomal dominant manner, or be associated with Williams-Beuren syndrome, a complex developmental disorder caused by a microdeletion of chromosome 7q11.23. ELN on 7q11.23, which encodes elastin, is the only known gene to be recurrently mutated in less than half of SVAS patients. METHODS: Whole-exome sequencing (WES) was performed for seven familial SVAS families to identify other causative gene mutations of SVAS. RESULTS: Three truncating mutations and three intragenic deletions affecting ELN were identified, yielding a diagnostic efficiency of 6/7 (85%). The deletions, which explained 3/7 of the present cohort, spanned 1-29 exons, which might be missed in the course of mutational analysis targeting point mutations. The presence of such deletions was validated by both WES-based copy number estimation and multiplex ligation-dependent probe amplification analyses, and their pathogenicity was reinforced by co-segregation with clinical presentations. CONCLUSIONS: The majority of familial SVAS patients appear to carry ELN mutations, which strongly indicates that elastin is the most important causative gene for SVAS. The frequency of intragenic deletions highlights the need for quantitative tests to analyze ELN for efficient genetic diagnosis of SVAS.

    DOI: 10.1016/j.ijcard.2018.09.032

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  401. Planetary-scale streak structure reproduced in high-resolution simulations of the Venus atmosphere with a low-stability layer Reviewed

    Hiroki Kashimura, Norihiko Sugimoto, Masahiro Takagi, Yoshihisa Matsuda, Wataru Ohfuchi, Takeshi Enomoto, Kensuke Nakajima, Masaki Ishiwatari, Takao M. Sato, George L. Hashimoto, Takehiko Satoh, Yoshiyuki O. Takahashi, Yoshi-Yuki Hayashi

    NATURE COMMUNICATIONS   Vol. 10 ( 1 ) page: 23   2019.1

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    Cloud patterns are important clues for revealing the atmospheric circulation of Venus. Recently, a planetary-scale streak structure has been discovered in middle- and lower-cloud images of Venus' night-side taken by IR2, the 2-mu m camera, on board the Akatsuki orbiter. However, its formation mechanism has not been investigated. Here we succeed, for the first time, in reproducing the patterns of the observed streak structure, as regions of strong downward flows that develop in high-resolution global simulations of the Venus atmosphere. The streaks are formed in both hemispheres with equatorial symmetry, which is caused by equatorial Rossby-like and Kelvin-like waves with zonal wavenumber one. The low-stability layer that has been suggested by past observations is essential for reproducing the streak structure. The streaks of downward flow result from the interaction of the meridionally tilted phase lines of the Rossby-like waves and the characteristics of baroclinic instability produced around the low-stability layer.

    DOI: 10.1038/s41467-018-07919-y

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  402. Investigation of appropriate semi-quantitative index for assessment of esophageal and breast cancer treatment response in Japanese patients using <sup>18</sup>F-FDG PET/CT findings. Reviewed

    Tsuchitani T, Takahashi Y, Maeda Y, Oda M, Enoki T, Kotoura N, Kitajima K

    Hellenic journal of nuclear medicine   Vol. 22 ( 1 ) page: 20 - 24   2019.1

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    DOI: 10.1967/s002449910955

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  403. A novel CUL4B splice site variant in a young male exhibiting less pronounced features. Reviewed International journal

    Yuji Nakamura, Yusuke Okuno, Hideki Muramatsu, Tomoko Kawai, Kazuhito Satou, Daisuke Ieda, Ikumi Hori, Kei Ohashi, Yutaka Negishi, Ayako Hattori, Yoshiyuki Takahashi, Seiji Kojima, Shinji Saitoh

    Human genome variation   Vol. 6   page: 43 - 43   2019

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    Patients with variants in CUL4B exhibit syndromic intellectual disability (MIM #300354). A seven-year-old boy presented with intellectual disability, a history of seizure, characteristic facial features, and short stature. Whole-exome sequencing detected a c.974+3A>G variant in CUL4B, which was subsequently confirmed to disrupt mRNA splicing. The current patient showed less pronounced phenotypic features compared with the previously reported cases. This report, therefore, provides evidence of genotype-phenotype correlations in CUL4B-related disorders.

    DOI: 10.1038/s41439-019-0074-6

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  404. [LOCALIZED AMYLOIDOSIS OF THE RENAL SINUS: A CASE REPORT].

    Ohashi T, Tanaka J, Asai K, Shimada S, Takahashi Y, Furukawa T

    Nihon Hinyokika Gakkai zasshi. The japanese journal of urology   Vol. 110 ( 3 ) page: 191 - 195   2019

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    DOI: 10.5980/jpnjurol.110.191

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  405. Ureteral dilatation detected in magnetic resonance imaging predicts vesicoureteral reflux in children with urinary tract infection. Reviewed International journal

    Norihiro Murakami, Jun-Ichi Kawada, Azumi Watanabe, Toshinao Arakawa, Takamasa Kano, Takako Suzuki, Ryo Tanaka, Daiei Kojima, Yoshihiko Kawano, Shin Hoshino, Hideki Muramatsu, Yoshiyuki Takahashi, Yoshiaki Sato, Masashi Koyama, Jun Natsume

    PloS one   Vol. 13 ( 12 ) page: e0209595   2018.12

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    OBJECTIVE: Urinary tract infection (UTI), one of the most common bacterial infections occurring during infancy and early childhood, is frequently associated with vesicoureteral reflux (VUR). Although several guidelines recommend performing ultrasonography as a screening test, its utility is not adequate and appropriate screening tests are strongly desirable. In this study, we evaluate the use of magnetic resonance imaging (MRI) as a screening test for VUR in children with UTI. METHODS: We prospectively studied 108 patients with suspected UTI between April 2014 and March 2016. UTI was diagnosed on the basis of diffusion-weighted MRI (DW-MRI) and urine culture findings. We measured ureteral dilatation using MRI in 96 patients with UTI and assessed the relationship between ureteral dilatation in MRI and VUR in 46 patients who underwent voiding cystourethrography (VCUG). RESULTS: Among 108 patients, 88 and 8 were diagnosed with upper and lower UTI, respectively. Among 46 patients who underwent VCUG, 23 had VUR (14 low grade and 9 high grade). Patients with ureteral dilatation detected on MRI had VUR more frequently than those without ureteral dilatation (any grades VUR, 71% vs. 32%; P = 0.02; high-grade VUR, 38% vs. 2%, P = 0.007). Overall, ureteral dilatation findings on MRI achieved sensitivity 65.2% and specificity 73.9% as a screening test for VUR. In addition, DW-MRI achieved sensitivity 100% and specificity 81.8% in the diagnosis of upper UTI. CONCLUSION: These findings suggested that MRI is a valuable tool for screening of VUR as well as diagnosis of upper UTI.

    DOI: 10.1371/journal.pone.0209595

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  406. Identification of potential pathogenic viruses in patients with acute myocarditis using next-generation sequencing. Reviewed International journal

    Suguru Takeuchi, Jun-Ichi Kawada, Yusuke Okuno, Kazuhiro Horiba, Takako Suzuki, Yuka Torii, Kazushi Yasuda, Atsushi Numaguchi, Taichi Kato, Yoshiyuki Takahashi, Yoshinori Ito

    Journal of medical virology   Vol. 90 ( 12 ) page: 1814 - 1821   2018.12

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    Myocarditis is an inflammatory disease of the myocardium and leads to cardiac dysfunction and heart failure. Although viral infections are considered to be the most common etiology of myocarditis, the identification of the causative virus is still challenging. Recently, next-generation sequencing (NGS) has been applied in the diagnosis of infectious diseases. The aim of the current study was to comprehensively analyze potential pathogenic microorganisms using NGS in the sera of patients with myocarditis. Twelve pediatric and five adult patients hospitalized for acute myocarditis were included. Serum samples in the acute phase were obtained and analyzed using NGS to detect pathogen-derived DNA and RNA. Viral sequence reads were detected in 7 (41%) of the 17 myocarditis patients by NGS. Among these patients, detection of Epstein-Barr virus, human parvovirus B19, torque teno virus, and respiratory syncytial virus reads by NGS was consistent with polymerase chain reaction or antigen test results in one patient each. A large number of human pegivirus reads were detected from one patient by RNA sequencing; however, its pathogenicity to human is unknown. Conversely, the number of detected virus-derived reads was small in most cases, and the pathophysiological role of these viruses remains to be clarified. No significant bacterial or fungal reads other than normal bacterial flora was detected. These data indicate that comprehensive detection of virus-derived DNA and RNA using NGS can be useful for the identification of potential pathogenic viruses in myocarditis.

    DOI: 10.1002/jmv.25263

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  407. Reduction of stratum corneum ceramides in Neu-Laxova syndrome caused by phosphoglycerate dehydrogenase deficiency. Reviewed International journal

    Takuya Takeichi, Yusuke Okuno, Akane Kawamoto, Takeshi Inoue, Eiko Nagamoto, Chiaki Murase, Eri Shimizu, Kenichi Tanaka, Yuichi Kageshita, Satoshi Fukushima, Michihiro Kono, Junko Ishikawa, Hironobu Ihn, Yoshiyuki Takahashi, Masashi Akiyama

    Journal of lipid research   Vol. 59 ( 12 ) page: 2413 - 2420   2018.12

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    Neu-Laxova syndrome (NLS) is a very rare autosomal recessive congenital disorder characterized by disturbed development of the central nervous system and the skin and caused by mutations in any of the three genes involved in de novo l-serine biosynthesis: PHGDH, PSAT1, and PSPH l-Serine is essential for the biosynthesis of phosphatidylserine and sphingolipids. The extracellular lipid of the stratum corneum, of which sphingolipid constitutes a significant part, plays a primary role in skin barrier function. Here, we describe a Japanese NLS pedigree with a previously unreported nonsense mutation in PHGDH and a unique inversion of chromosome 1. In addition, the levels of 11 major ceramide classes in the tape-stripped stratum corneum of the NLS patient's skin were assessed by LC/MS. Notably, lower amounts of ceramides of all classes were found in the patient's stratum corneum than in those of controls. This is the first report to demonstrate the reduction of ceramides in the stratum corneum of an NLS patient due to PHGDH mutations. The clinical findings and a detailed analysis of ceramides from the stratum corneum in the family extend the spectrum of clinical anomalies and give us a clue to the pathomechanisms of ichthyosis in NLS patients with phosphoglycerate dehydrogenase deficiency.

    DOI: 10.1194/jlr.P087536

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  408. The presence of defective Epstein-Barr virus (EBV) infection in EBV-associated hematological malignancies

    Okuno, Y; Murata, T; Sato, Y; Ito, Y; Yoshida, K; Sawada, A; Shiraishi, Y; Miyano, S; Takahashi, Y; Kojima, S; Ogawa, S; Kimura, H

    CANCER SCIENCE   Vol. 109   page: 642 - 642   2018.12

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  409. The presence of defective Epstein-Barr virus (EBV) infection in EBV-associated hematological malignancies

    Okuno Yusuke, Murata Takayuki, Sato Yoshitaka, Ito Yoshinori, Yoshida Kenichi, Sawada Akihisa, Shiraishi Yuichi, Miyano Satoru, Takahashi Yoshiyuki, Kojima Seiji, Ogawa Seishi, Kimura Hiroshi

    CANCER SCIENCE   Vol. 109   page: 642-642   2018.12

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  410. Clinical and Genetic Characteristics of Patients with Shwachman-Diamond Syndrome in Japan

    Watanabe Kenichiro, Kanegane Hirokazu, Hamabata Takayuki, Kozuki Kagehiro, Umeda Katsutsugu, Ueno Hiroo, Yoshida Kenichi, Hama Asahito, Okuno Yusuke, Muramatsu Hideki, Takahashi Yoshiyuki, Hasegawa Daisuke, Manabe Atsushi, Ohara Akira, Ito Masafumi, Ogawa Seishi, Kojima Seiji, Ito Etsuro

    BLOOD   Vol. 132   2018.11

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    DOI: 10.1182/blood-2018-99-114313

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  411. Combination of TREC Measurement and Next-Generation Sequencing in Newborn Screening for Severe Combined Immunodeficiency: A Pilot Program in Japan

    Muramatsu Hideki, Kojima Daiei, Okuno Yusuke, Kataoka Shinsuke, Nakajima Yoko, Ito Tetsuya, Tsuge Ikuya, Yoshimi Sakai, Kato Tomoaki, Kojima Seiji, Takahashi Yoshiyuki

    BLOOD   Vol. 132   2018.11

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    DOI: 10.1182/blood-2018-99-118261

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  412. <i>Piggybac</i> CD19 CAR T Cells Eradicate CNS Leukemia By Direct Delivery into Cerebral Ventricle of Xenograft Mice Model

    Tanaka, K; Kato, I; Tanaka, M; Morita, D; Takahashi, Y; Nakahata, T; Umeda, K; Hiramatsu, H; Adachi, S; Takita, J; Nakazawa, Y

    BLOOD   Vol. 132   2018.11

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    DOI: 10.1182/blood-2018-99-111733

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  413. Randomized Trial of Two Dosages of Rabbit Antithymocyte Globulin in Patients with Aplastic Anemia

    Narita, A; Zhu, XF; Muramatsu, H; Chen, XJ; Guo, Y; Yang, WY; Zhang, JL; Liu, F; Jang, JH; Kook, H; Kim, H; Usuki, K; Yamazaki, H; Takahashi, Y; Nakao, S; Lee, JW; Kojima, S

    BLOOD   Vol. 132   2018.11

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    DOI: 10.1182/blood-2018-99-111323

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  414. The Presence of Defective Epstein-Barr Virus (EBV) Infection in Patients with EBV-Associated Hematological Malignancy

    Okuno Yusuke, Murata Takayuki, Sato Yoshitaka, Muramatsu Hideki, Ito Yoshinori, Watanabe Takahiro, Okuno Tatsuya, Murakami Norihiro, Yoshida Kenichi, Sawada Akihisa, Inoue Masami, Kawa Keisei, Seto Masao, Ohshima Koichi, Shiraishi Yuichi, Chiba Kenichi, Tanaka Hiroko, Miyano Satoru, Narita Yohei, Yoshida Masahiro, Goshima Fumi, Kawada Junichi, Nishida Tetsuya, Kiyoi Hitoshi, Kato Seiichi, Nakamura Shigeo, Morishima Satoko, Fujiwara Shigeyoshi, Shimizu Norio, Isobe Yasushi, Noguchi Masaaki, Kikuta Atsushi, Iwatsuki Keiji, Takahashi Yoshiyuki, Kojima Seiji, Ogawa Seishi, Kimura Hiroshi

    BLOOD   Vol. 132   2018.11

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    DOI: 10.1182/blood-2018-99-113801

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  415. A Case of Dyskeratosis Congenital who Showed the Improvement of Telomere Length After Receiving Androgens for 11 Years

    Wakamatsu Manabu, Nishio Nobuhiro, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Ichikawa Daisuke, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S65 - S65   2018.11

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  416. A Case of MLL-AF9 Fusion Positive Malignant Lymphoma Without Bone Marrow Infiltration Either at First Diagnosis or at Relapse

    Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Nishikawa Eri, Nishio Nobuhiro, Okuno Yusuke, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S99 - S99   2018.11

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  417. Application of RNA Sequencing for Early Diagnosis of NUT Carcinoma: A Case Report

    Miwata Shunsuke, Narita Atsushi, Wakamatsu Manabu, Narita Kotaro, Kitazawa Hironobu, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Taniguchi Rieko, Ichikawa Daisuke, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Nishio Nobuhiro, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S74 - S74   2018.11

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  418. A Case of Thoracic Tumor Caused by SMARCA4 Biallelic Inactivation

    Ichikawa Daisuke, Muramatsu Hideki, Okuno Yusuke, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Hamada Motoharu, Kataoka Shinsuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S74 - S74   2018.11

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  419. Diagnosis of Inherited Bone Marrow Failure Syndromes Using Sequencing Approaches

    Narita, A; Muramatsu, H; Okuno, Y; Yoshida, K; Shiraishi, Y; Sakaguchi, H; Kawashima, N; Wang, XA; Xu, YY; Chiba, K; Tanaka, H; Hama, A; Sanada, M; Hitoshi, K; Yamaguchi, H; Ohga, S; Manabe, A; Harigae, H; Kunishima, S; Ishii, E; Kobayashi, M; Koike, K; Watanabe, K; Ito, E; Takata, M; Yabe, M; Ogawa, S; Miyano, S; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S7 - S7   2018.11

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  420. Disappearance of Minimal Residual Disease Demonstrates Graft Versus Neuroblastoma Effect After KIR-Ligand Mismatched Allogeneic Cord Blood Transplantation

    Nishio, N; Kataoka, S; Miwata, S; Narita, K; Kitazawa, H; Hamada, M; Ichikawa, D; Taniguchi, R; Murakami, N; Suzuki, K; Kawashima, N; Nishikawa, E; Okuno, Y; Narita, A; Muramatsu, H; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S8 - S8   2018.11

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  421. Herpesvirus Infection in Pediatric Autologous Hematopoietic Stem Cell Transplantation

    Miura, H; Kudo, K; Tanaka, M; Takahashi, Y; Kojima, S; Yoshikawa, T

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S60 - S61   2018.11

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  422. Unmet Medical Needs for Japanese Kampo Medicine in Children with Hematological and Oncological Disease

    Kawashima, N; Miwata, S; Narita, K; Kataoka, S; Hamada, M; Murakami, N; Ichikawa, D; Suzuki, K; Kitazawa, H; Nishikawa, E; Okuno, Y; Taniguchi, R; Narita, A; Muramatsu, H; Nishio, N; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S33 - S33   2018.11

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  423. Urgent Successful HLA-Haploidentical Stem Cell Transplantation in Children with Aplastic Anemia Considering Upfront SCT Strategy

    Nishikawa, E; Wakamatsu, M; Miwata, S; Narita, K; Murakami, N; Kataoka, S; Hamada, M; Ichikawa, D; Kitazawa, H; Taniguchi, R; Suzuki, K; Kawashima, N; Okuno, Y; Narita, A; Muramatsu, H; Nishio, N; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S65 - S66   2018.11

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  424. Morphological Features of Bone Marrow in Patients with Inherited Bone Marrow Failure

    Hama, A; Hasegawa, D; Manabe, A; Nozawa, K; Narita, A; Okuno, Y; Muramatsu, H; Takahashi, Y; Watanabe, K; Ohara, A; Ito, M; Kojima, S

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S66 - S66   2018.11

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  425. Relapse After Spontaneous Molecular Remission in a Female with Congenital Acute Myeloid Leukemia with KAT6A-EP300 Fusion Transcript

    Taniguchi, R; Kawashima, N; Ikawa, Y; Wakamatsu, M; Miwata, S; Narita, K; Kataoka, S; Hamada, M; Murakami, N; Ichikawa, D; Kitazawa, H; Suzuki, K; Nishikawa, E; Narita, A; Okuno, Y; Muramatsu, H; Nishio, N; Nishimura, R; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S98 - S98   2018.11

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  426. NGS-Based Methylation Analysis of Juvenile Myelomonocytic Leukemia using the Digital Restriction Enzyme Analysis of Methylation (DREAM) Method

    Kitazawa, H; Murakami, N; Okuno, Y; Muramatsu, H; Wakamatsu, M; Miwata, S; Narita, K; Kataoka, S; Hamada, M; Ichikawa, D; Taniguchi, R; Suzuki, K; Kawashima, N; Nishikawa, E; Narita, A; Nishio, N; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S22 - S23   2018.11

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  427. Urgent Successful HLA-Haploidentical Stem Cell Transplantation in Children with Aplastic Anemia Considering Upfront SCT Strategy

    Nishikawa Eri, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Murakami Norihiro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Kitazawa Hironobu, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S65-S66   2018.11

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  428. Relapse After Spontaneous Molecular Remission in a Female with Congenital Acute Myeloid Leukemia with KAT6A-EP300 Fusion Transcript

    Taniguchi Rieko, Kawashima Nozomu, Ikawa Yasuhiro, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Ichikawa Daisuke, Kitazawa Hironobu, Suzuki Kyogo, Nishikawa Eri, Narita Atsushi, Okuno Yusuke, Muramatsu Hideki, Nishio Nobuhiro, Nishimura Ryosei, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S98-S98   2018.11

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  429. Unmet Medical Needs for Japanese Kampo Medicine in Children with Hematological and Oncological Disease

    Kawashima Nozomu, Miwata Shunsuke, Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Ichikawa Daisuke, Suzuki Kyogo, Kitazawa Hironobu, Nishikawa Eri, Okuno Yusuke, Taniguchi Rieko, Narita Atsushi, Muramatsu Hideki, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S33-S33   2018.11

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  430. NGS-Based Methylation Analysis of Juvenile Myelomonocytic Leukemia using the Digital Restriction Enzyme Analysis of Methylation (DREAM) Method

    Kitazawa Hironobu, Murakami Norihiro, Okuno Yusuke, Muramatsu Hideki, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S22-S23   2018.11

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  431. Morphological Features of Bone Marrow in Patients with Inherited Bone Marrow Failure

    Hama Asahito, Hasegawa Daisuke, Manabe Atsushi, Nozawa Kazue, Narita Atsushi, Okuno Yusuke, Muramatsu Hideki, Takahashi Yoshiyuki, Watanabe Kenichiro, Ohara Akira, Ito Masafumi, Kojima Seiji

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S66-S66   2018.11

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  432. Disappearance of Minimal Residual Disease Demonstrates Graft Versus Neuroblastoma Effect After KIR-Ligand Mismatched Allogeneic Cord Blood Transplantation

    Nishio Nobuhiro, Kataoka Shinsuke, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S8-S8   2018.11

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  433. A Case of Dyskeratosis Congenital who Showed the Improvement of Telomere Length After Receiving Androgens for 11 Years

    Wakamatsu Manabu, Nishio Nobuhiro, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Ichikawa Daisuke, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S65-S65   2018.11

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  434. A Case of MLL-AF9 Fusion Positive Malignant Lymphoma Without Bone Marrow Infiltration Either at First Diagnosis or at Relapse

    Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Nishikawa Eri, Nishio Nobuhiro, Okuno Yusuke, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S99-S99   2018.11

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  435. A Case of Thoracic Tumor Caused by SMARCA4 Biallelic Inactivation

    Ichikawa Daisuke, Muramatsu Hideki, Okuno Yusuke, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Hamada Motoharu, Kataoka Shinsuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S74-S74   2018.11

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  436. Application of RNA Sequencing for Early Diagnosis of NUT Carcinoma: A Case Report

    Miwata Shunsuke, Narita Atsushi, Wakamatsu Manabu, Narita Kotaro, Kitazawa Hironobu, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Taniguchi Rieko, Ichikawa Daisuke, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Nishio Nobuhiro, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S74-S74   2018.11

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  437. Compound heterozygous RYR1 mutations by whole exome sequencing in a family with three repeated affected fetuses with fetal akinesia. Reviewed

    Suzumori N, Inagaki H, Ohtani A, Kumagai K, Takeda E, Yoshihara H, Sawada Y, Inuzuka S, Iwagaki S, Takahashi Y, Kurahashi H, Sugiura-Ogasawara M

    European journal of obstetrics, gynecology, and reproductive biology   Vol. 230   page: 200 - 202   2018.11

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    DOI: 10.1016/j.ejogrb.2018.09.013

    PubMed

  438. Diagnosis of Inherited Bone Marrow Failure Syndromes Using Sequencing Approaches

    Narita Atsushi, Muramatsu Hideki, Okuno Yusuke, Yoshida Kenichi, Shiraishi Yuichi, Sakaguchi Hirotoshi, Kawashima Nozomu, Wang Xinan, Xu Yinyan, Chiba Kenichi, Tanaka Hiroko, Hama Asahito, Sanada Masashi, Hitoshi Kanno, Yamaguchi Hiroki, Ohga Shouichi, Manabe Atsushi, Harigae Hideo, Kunishima Shinji, Ishii Eiichi, Kobayashi Masao, Koike Kenichi, Watanabe Kenichiro, Ito Etsuro, Takata Minoru, Yabe Miharu, Ogawa Seishi, Miyano Satoru, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 65   page: S7-S7   2018.11

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  439. 骨髄MRDの消失は神経芽腫に対するKIRリガンド不一致同種臍帯血移植後の移植片対腫瘍効果を示唆する(Disappearance of minimal residual disease demonstrates graft versus neuroblastoma effect after KIR-ligand mismatched allogeneic cord blood transplantation)

    Nishio Nobuhiro, Kataoka Shinsuke, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 154 - 154   2018.10

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  440. 自然寛解後に再発をきたしたKAT6A-EP300融合転写を伴う先天性急性骨髄性白血病(Relapse after spontaneous molecular remission in a female with congenital acute myeloid leukemia with KAT6A-EP300 fusion transcript)

    Taniguchi Rieko, Kawashima Nozomu, Ikawa Yasuhiro, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Ichikawa Daisuke, Kitazawa Hironobu, Suzuki Kyogo, Nishikawa Eri, Narita Atsushi, Okuno Yusuke, Muramatsu Hideki, Nishio Nobuhiro, Nishimura Ryosei, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 314 - 314   2018.10

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  441. 蛋白同化ステロイド内服を11年継続している先天性角化不全症の一例(A case of dyskeratosis congenital who showed the improvement of telomere length after receiving androgens for 11 years)

    Wakamatsu Manabu, Nishio Nobuhiro, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Ichikawa Daisuke, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 246 - 246   2018.10

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  442. 小児再生不良性貧血に対するUpfront移植を含むハプロ移植の7例(Urgent successful HLA-haploidentical stem cell transplantation in children with aplastic anemia considering upfront SCT strategy)

    Nishikawa Eri, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Murakami Norihiro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Kitazawa Hironobu, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 247 - 247   2018.10

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  443. 初発時および再発時に骨髄浸潤を認めなかったMLL/AF9融合遺伝子を持つ悪性リンパ腫の1例(A case of MLL-AF9 fusion positive malignant lymphoma without bone marrow infiltration either at first diagnosis or at relapse)

    Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Nishikawa Eri, Nishio Nobuhiro, Okuno Yusuke, Kawashima Nozomu, Narita Atsushi, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 316 - 316   2018.10

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  444. 小児がん拠点病院単一施設における漢方診療の患者ニーズ調査(Unmet medical needs for Japanese Kampo medicine in children with hematological and oncological disease)

    Kawashima Nozomu, Miwata Shunsuke, Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Ichikawa Daisuke, Suzuki Kyogo, Kitazawa Hironobu, Nishikawa Eri, Okuno Yusuke, Taniguchi Rieko, Narita Atsushi, Muramatsu Hideki, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 219 - 219   2018.10

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  445. シーケンスによる先天性骨髄不全症の診断(Diagnosis of Inherited Bone Marrow Failure Syndromes Using Sequencing Approaches)

    Narita Atsushi, Muramatsu Hideki, Okuno Yusuke, Yoshida Kenichi, Shiraishi Yuichi, Sakaguchi Hirotoshi, Kawashima Nozomu, Wang Xinan, Xu Yinyan, Chiba Kenichi, Tanaka Hiroko, Hama Asahito, Sanada Masashi, Kanno Hitoshi, Yamaguchi Hiroki, Ohga Shouichi, Manabe Atsushi, Harigae Hideo, Kunishima Shinji, Ishii Eiichi, Kobayashi Masao, Koike Kenichi, Watanabe Kenichiro, Ito Etsuro, Takata Minoru, Yabe Miharu, Ogawa Seishi, Miyano Satoru, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 153 - 153   2018.10

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  446. RNAシーケンスが早期診断に有用であったNUT carcinomaの1例(Application of RNA sequencing for early diagnosis of NUT carcinoma: a case report)

    Miwata Shunsuke, Narita Atsushi, Wakamatsu Manabu, Narita Kotaro, Kitazawa Hironobu, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Taniguchi Rieko, Ichikawa Daisuke, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Nishio Nobuhiro, Muramatsu Hideki, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 255 - 255   2018.10

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  447. SMARCA4の不活化変異により発症した胸部腫瘍の1例(A case of thoracic tumor caused by SMARCA4 biallelic inactivation)

    Ichikawa Daisuke, Muramatsu Hideki, Okuno Yusuke, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Hamada Motoharu, Kataoka Shinsuke, Taniguchi Rieko, Murakami Norihiro, Suzuki Kyogo, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 255 - 255   2018.10

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  448. DREAM法を用いた若年性骨髄単球性白血病に対するメチル化解析(NGS-based methylation analysis of juvenile myelomonocytic leukemia using the digital restriction enzyme analysis of methylation(DREAM) method)

    Kitazawa Hironobu, Murakami Norihiro, Okuno Yusuke, Muramatsu Hideki, Wakamatsu Manabu, Miwata Shunsuke, Narita Kotaro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Narita Atsushi, Nishio Nobuhiro, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 206 - 206   2018.10

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  449. Defibrotide for Prophylaxis of Hepatic Veno-Occlusive Disease Hematopoietic Stem Cell Transplantation: Results from the Japanese Prophylactic Use, Phase II, Randomized Trial

    Kikuta, A; Fukuda, T; Ohashi, K; Taniguchi, S; Asano-Mori, Y; Horibe, K; Goto, H; Nakamae, H; Ogawa, H; Tamaki, H; Takahashi, Y; Kimura, T; Yoshimura, K; Umemura, K; Ogawa, C

    BONE MARROW TRANSPLANTATION   Vol. 53   page: 316 - 317   2018.9

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    Web of Science

  450. Defibrotide for Treatment of Hepatic Veno-Occlusive Disease Following Hematopoietic Stem Cell Transplantation: Results from the Japanese Therapeutic Use Protocol

    Kikuta, A; Fukuda, T; Ohashi, K; Taniguchi, S; Asano-Mori, Y; Horibe, K; Goto, H; Nakamae, H; Ogawa, H; Tamaki, H; Takahashi, Y; Kimura, T; Yoshimura, K; Umemura, K; Ogawa, C

    BONE MARROW TRANSPLANTATION   Vol. 53   page: 318 - 319   2018.9

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  451. Updated Treatment Algorism for Children with Acquired Aplastic Anemia: A Report from the Japan Childhood Aplastic Anemia Study Group

    Yoshida, N; Narita, A; Muramatsu, H; Kobayashi, R; Yabe, H; Kudo, K; Yagasaki, H; Watanabe, K; Morimoto, A; Ito, E; Ohga, S; Ohara, A; Takahashi, Y; Kojima, S

    BONE MARROW TRANSPLANTATION   Vol. 53   page: 34 - 35   2018.9

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  452. Whole-exome analysis to detect congenital hemolytic anemia mimicking congenital dyserythropoietic anemia. Reviewed

    Motoharu Hamada, Sayoko Doisaki, Yusuke Okuno, Hideki Muramatsu, Asahito Hama, Nozomu Kawashima, Atsushi Narita, Nobuhiro Nishio, Kenichi Yoshida, Hitoshi Kanno, Atsushi Manabe, Takashi Taga, Yoshiyuki Takahashi, Satoru Miyano, Seishi Ogawa, Seiji Kojima

    International journal of hematology   Vol. 108 ( 3 ) page: 306 - 311   2018.9

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    Congenital dyserythropoietic anemia (CDA) is a heterogeneous group of rare congenital disorders characterized by ineffective erythropoiesis and dysplastic changes in erythroblasts. Diagnosis of CDA is based primarily on the morphology of bone marrow erythroblasts; however, genetic tests have recently become more important. Here, we performed genetic analysis of 10 Japanese patients who had been diagnosed with CDA based on laboratory findings and morphological characteristics. We examined 10 CDA patients via central review of bone marrow morphology and genetic analysis for congenital bone marrow failure syndromes. Sanger sequencing for CDAN1, SEC23B, and KLF1 was performed for all patients. We performed whole-exome sequencing in patients without mutation in these genes. Three patients carried pathogenic CDAN1 mutations, whereas no SEC23B mutations were identified in our cohort. WES unexpectedly identified gene mutations known to cause congenital hemolytic anemia in two patients: canonical G6PD p.Val394Leu mutation and SPTA1 p.Arg28His mutation. Comprehensive genetic analysis is warranted for more effective diagnosis of patients with suspected CDA.

    DOI: 10.1007/s12185-018-2482-7

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    PubMed

  453. Risk Factors for Viral Infections in Pediatric Patients Undergoing Allogeneic Hematopoietic Stem Cell Transplantation

    Narita, A; Ito, Y; Torii, Y; Kawada, JI; Miwata, S; Narita, K; Kitazawa, H; Hamada, M; Kataoka, S; Ichikawa, D; Ito, R; Murakami, N; Kojima, D; Suzuki, K; Nishikawa, E; Kawashima, N; Okuno, Y; Muramatsu, H; Hama, A; Kojima, S; Takahashi, Y

    BONE MARROW TRANSPLANTATION   Vol. 53   page: 588 - 589   2018.9

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  454. Serum anti-<i>Helicobacter pylori</i> antibody titer and its association with gastric nodularity, atrophy, and age: A cross-sectional study. Reviewed International journal

    Toyoshima O, Nishizawa T, Sakitani K, Yamakawa T, Takahashi Y, Yamamichi N, Hata K, Seto Y, Koike K, Watanabe H, Suzuki H

    World journal of gastroenterology   Vol. 24 ( 35 ) page: 4061 - 4068   2018.9

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    AIM: To clarify the role of serum anti-Helicobacter pylori (H. pylori) antibody titers in gastric cancer. METHODS: In this cross-sectional study, the effect of patients' baseline characteristics and endoscopic findings on their serum antibody titers were assessed. We evaluated consecutive patients who underwent esophagogastroduodenoscopy and their first evaluation for H. pylori infection using a serum antibody test. We excluded patients with a history of eradication therapy. The participants were divided into four groups according to their E-plate serum antibody titer. Patients with serum antibody titers < 3, 3-9.9, 10-49.9, and ≥ 50 U/mL were classified into groups A, B, C, and D, respectively. RESULTS: In total, 874 participants were analyzed with 70%, 16%, 8.7%, and 5.1% of them in the groups A, B, C, and D, respectively. Patients in group C were older than patients in groups A and B. Gastric open-type atrophy, intestinal metaplasia, enlarged folds, diffuse redness, and duodenal ulcers were associated with a high titer. Regular arrangements of collecting venules, fundic gland polyps, superficial gastritis, and gastroesophageal reflux disease were related to a low titer. Multivariate analysis revealed that nodularity (P = 0.0094), atrophy (P = 0.0076), and age 40-59 years (vs age ≥ 60 years, P = 0.0090) were correlated with a high serum antibody titer in H. pylori-infected patients. Intestinal metaplasia and atrophy were related to age ≥ 60 years in group C and D. CONCLUSION: Serum antibody titer changes with age, reflects gastric mucosal inflammation, and is useful in predicting the risk of gastric cancer.

    DOI: 10.3748/wjg.v24.i35.4061

    PubMed

  455. Risk Factors for Viral Infections in Pediatric Patients Undergoing Allogeneic Hematopoietic Stem Cell Transplantation

    Narita Atsushi, Ito Yoshinori, Torii Yuka, Kawada Jun-Ichi, Miwata Shunsuke, Narita Kotaro, Kitazawa Hironobu, Hamada Motoharu, Kataoka Shinsuke, Ichikawa Daisuke, Ito Rieko, Murakami Norihiro, Kojima Daiei, Suzuki Kyogo, Nishikawa Eri, Kawashima Nozomu, Okuno Yusuke, Muramatsu Hideki, Hama Asahito, Kojima Seiji, Takahashi Yoshiyuki

    BONE MARROW TRANSPLANTATION   Vol. 53   page: 588-589   2018.9

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  456. Integration Mapping of piggyBac-Mediated CD19 Chimeric Antigen Receptor T Cells Analyzed by Novel Tagmentation-Assisted PCR. Reviewed International journal

    Motoharu Hamada, Nobuhiro Nishio, Yusuke Okuno, Satoshi Suzuki, Nozomu Kawashima, Hideki Muramatsu, Shoma Tsubota, Matthew H Wilson, Daisuke Morita, Shinsuke Kataoka, Daisuke Ichikawa, Norihiro Murakami, Rieko Taniguchi, Kyogo Suzuki, Daiei Kojima, Yuko Sekiya, Eri Nishikawa, Atsushi Narita, Asahito Hama, Seiji Kojima, Yozo Nakazawa, Yoshiyuki Takahashi

    EBioMedicine   Vol. 34   page: 18 - 26   2018.8

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    Insertional mutagenesis is an important risk with all genetically modified cell therapies, including chimeric antigen receptor (CAR)-T cell therapy used for hematological malignancies. Here we describe a new tagmentation-assisted PCR (tag-PCR) system that can determine the integration sites of transgenes without using restriction enzyme digestion (which can potentially bias the detection) and allows library preparation in fewer steps than with other methods. Using this system, we compared the integration sites of CD19-specific CAR genes in final T cell products generated by retrovirus-based and lentivirus-based gene transfer and by the piggyBac transposon system. The piggyBac system demonstrated lower preference than the retroviral system for integration near transcriptional start sites and CpG islands and higher preference than the lentiviral system for integration into genomic safe harbors. Integration into or near proto-oncogenes was similar in all three systems. Tag-PCR mapping is a useful technique for assessing the risk of insertional mutagenesis.

    DOI: 10.1016/j.ebiom.2018.07.008

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    PubMed

  457. Late-phase human herpesvirus 6B reactivation in hematopoietic stem cell transplant recipients. Reviewed International journal

    Hiroki Miura, Yoshiki Kawamura, Fumihiko Hattori, Makito Tanaka, Kazuko Kudo, Masaru Ihira, Hiroshi Yatsuya, Yoshiyuki Takahashi, Seiji Kojima, Tetsushi Yoshikawa

    Transplant infectious disease : an official journal of the Transplantation Society   Vol. 20 ( 4 ) page: e12916   2018.8

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    BACKGROUND: We sought to determine whether late-phase human herpesvirus 6B (HHV-6B) infection in hematopoietic stem cell transplant (HSCT) recipients was associated with serious outcomes and mortality. METHODS: The occurrence and course of HHV-6B infection was monitored for at least 60 days after transplant using virus isolation and real-time polymerase chain reaction. Risk factors for late-phase HHV-6B infection were examined, and the propensity score was calculated with significant risk factors. The inverse probability-weighted multivariable logistic regression analysis was performed to estimate odds ratios (ORs) and the 95% confidence intervals (95% CI) for mortality. RESULTS: Late-phase HHV-6B infection was observed in 12/89 (13.5%) of the HSCT recipients. Older age (OR: 10.3, 95% CI: 2.1/72.9, P = .0027), hematologic malignancy (OR: 10.3, 95% CI: 1.8/97.1, P = .0063), unrelated donor transplantation (OR: 5.3, 95% CI: 1.1/36.0, P = .0345), and sex-mismatched donor transplantation (OR: 6.3, 95% CI: 1.4/39.5, P = .0149) were identified as risk factors for late-phase HHV-6B infection. Fifteen subjects died (17%). Inverse probability-weighted multivariable logistic model analysis revealed that late-phase HHV-6B infection was an independent risk factor for mortality (OR: 4.2, 95% CI: 1.7/11.0, P = .0012). Among 5 of the fatal cases of late-phase HHV-6B infection, viral infection might be associated with severe clinical manifestations. CONCLUSION: Late-phase HHV-6B infection in HSCT recipients was associated with worse outcomes. The full spectrum of clinical features of the infection has not been fully elucidated, and therefore, recipients with high-risk factors for late-phase HHV-6B infection should be carefully monitored.

    DOI: 10.1111/tid.12916

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    PubMed

  458. Expression and localization of aquaporins 3 and 7 in bull spermatozoa and their relevance to sperm motility after cryopreservation. Reviewed

    Fujii T, Hirayama H, Fukuda S, Kageyama S, Naito A, Yoshino H, Moriyasu S, Yamazaki T, Sakamoto K, Hayakawa H, Takahashi K, Takahashi Y, Sawai K

    The Journal of reproduction and development   Vol. 64 ( 4 ) page: 327 - 335   2018.8

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    Artificial insemination with cryopreserved semen is a well-developed technique commonly used for controlled reproduction in cattle. However, despite current technical advances, cryopreservation continues to damage bull spermatozoa, resulting in a loss of approximately 30 to 50% of viable spermatozoa post thawing. To further improve the efficiency of cryopreservation of bull spermatozoa, understanding the molecular mechanisms underlying the cryobiological properties that affect cryoinjuries during cryopreservation process of bull spermatozoa is required. In this study, we examined the expression and localization of aquaporin (AQP) 3 and AQP7 in fresh, cooled, and frozen-thawed bull spermatozoa. Furthermore, we investigated the relevance of AQP3 and AQP7 to motility and to membrane integrity in frozen-thawed bull spermatozoa. Western blotting against AQP3 and AQP7 in bull spermatozoa revealed bands with molecular weights of approximately 42 kDa and 53 kDa, respectively. In immunocytochemistry analyses, immunostaining of AQP3 was clearly observed in the principal piece of the sperm tail. Two immunostaining patterns were observed for AQP7 -pattern 1: diffuse staining in head and entire tail, and pattern 2: diffuse staining in head and clear staining in mid-piece. Cooling and freeze-thawing did not affect the localization pattern of AQP7 and the relative abundances of AQP3 and AQP7 evaluated by Western blotting. Furthermore, we demonstrated that the relative abundances of AQP3 and AQP7 varied among ejaculates, and they were positively related to sperm motility, particularly sperm velocity, post freeze-thawing. Our findings suggest that AQP3 and AQP7 are possibly involved in the tolerance to freeze-thawing in bull spermatozoa, particularly in the sperm's tail.

    DOI: 10.1262/jrd.2017-166

    PubMed

  459. 小児循環器疾患の基礎研究から臨床への応用 家族性大動脈弁上狭窄において、遺伝子内微小欠失の頻度は高い 次世代シーケンサーによる7家系の解析と臨床応用への可能性

    早野 聡, 奥野 友介, 堤 真紀子, 稲垣 秀人, 深澤 佳絵, 倉橋 浩樹, 小島 勢二, 高橋 義行, 加藤 太一

    日本小児循環器学会雑誌   Vol. 34 ( Suppl.1 ) page: s1 - 122   2018.7

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  460. 冠動脈病変合併リスクの高い川崎病患者に対する投与期間を短縮した免疫グロブリン・プレドニゾロン初期併用療法 多施設共同ランダム化比較試験による検証

    加藤 太一, 深澤 佳絵, 山本 英範, 沼口 敦, 早野 聡, 岸本 泰明, 長井 典子, 安藤 昌彦, 高橋 義行

    日本小児循環器学会雑誌   Vol. 34 ( Suppl.1 ) page: s1 - 283   2018.7

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  461. Diagnostic performance of 18F-FDG PET/CT and whole-body diffusion-weighted imaging with background body suppression (DWIBS) in detection of lymph node and bone metastases from pediatric neuroblastoma. Reviewed

    Hiroaki Ishiguchi, Shinji Ito, Katsuhiko Kato, Yusuke Sakurai, Hisashi Kawai, Naotoshi Fujita, Shinji Abe, Atsushi Narita, Nobuhiro Nishio, Hideki Muramatsu, Yoshiyuki Takahashi, Shinji Naganawa

    Annals of nuclear medicine   Vol. 32 ( 5 ) page: 348 - 362   2018.6

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    OBJECTIVE: Recent many studies have shown that whole body "diffusion-weighted imaging with background body signal suppression" (DWIBS) seems a beneficial tool having higher tumor detection sensitivity without ionizing radiation exposure for pediatric tumors. In this study, we evaluated the diagnostic performance of whole body DWIBS and 18F-FDG PET/CT for detecting lymph node and bone metastases in pediatric patients with neuroblastoma. METHODS: Subjects in this retrospective study comprised 13 consecutive pediatric patients with neuroblastoma (7 males, 6 females; mean age, 2.9 ± 2.0 years old) who underwent both 18F-FDG PET/CT and whole-body DWIBS. All patients were diagnosed as neuroblastoma on the basis of pathological findings. Eight regions of lymph nodes and 17 segments of skeletons in all patients were evaluated. The images of 123I-MIBG scintigraphy/SPECT-CT, bone scintigraphy/SPECT, and CT were used to confirm the presence of lymph node and bone metastases. Two radiologists trained in nuclear medicine evaluated independently the uptake of lesions in 18F-FDG PET/CT and the signal-intensity of lesions in whole-body DWIBS visually. Interobserver difference was overcome through discussion to reach a consensus. The sensitivities, specificities, and overall accuracies of 18F-FDG PET/CT and whole-body DWIBS were compared using McNemer's test. Positive predictive values (PPVs) and negative predictive values (NPVs) of both modalities were compared using Fisher's exact test. RESULTS: The total numbers of lymph node regions and bone segments which were confirmed to have metastasis in the total 13 patients were 19 and 75, respectively. The sensitivity, specificity, overall accuracy, PPV, and NPV of 18F-FDG PET/CT for detecting lymph node metastasis from pediatric neuroblastoma were 100, 98.7, 98.9, 95.0, and 100%, respectively, and those for detecting bone metastasis were 90.7, 73.1, 80.3, 70.1, and 91.9%, respectively. In contrast, the sensitivity, specificity, overall accuracy, PPV, and NPV of whole-body DWIBS for detecting bone metastasis from pediatric neuroblastoma were 94.7, 24.0, 53.0, 46.4 and 86.7%, respectively, whereas those for detecting lymph node metastasis were 94.7, 85.3, 87.2, 62.1, and 98.5%, respectively. The low specificity, overall accuracy, and PPV of whole-body DWIBS for detecting bone metastasis were due to a high incidence of false-positive findings (82/108, 75.9%). The specificity, overall accuracy, and PPV of whole-body DWIBS for detecting lymph node metastasis were also significantly lower than those of 18F-FDG PET/CT for detecting lymph node metastasis, although the difference between these 2 modalities was less than that for detecting bone metastasis. CONCLUSION: The specificity, overall accuracy, and PPV of whole-body DWIBS are significantly lower than those of 18F-FDG PET/CT because of a high incidence of false-positive findings particularly for detecting bone metastasis, whereas whole-body DWIBS shows a similar level of sensitivities for detecting lymph node and bone metastases to those of 18F-FDG PET/CT. DWIBS should be carefully used for cancer staging in children because of its high incidence of false-positive findings in skeletons.

    DOI: 10.1007/s12149-018-1254-z

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  462. Integrated molecular profiling of juvenile myelomonocytic leukemia. Reviewed International journal

    Norihiro Murakami, Yusuke Okuno, Kenichi Yoshida, Yuichi Shiraishi, Genta Nagae, Kyogo Suzuki, Atsushi Narita, Hirotoshi Sakaguchi, Nozomu Kawashima, Xinan Wang, Yinyan Xu, Kenichi Chiba, Hiroko Tanaka, Asahito Hama, Masashi Sanada, Masafumi Ito, Masashi Hirayama, Arata Watanabe, Toshihide Ueno, Seiji Kojima, Hiroyuki Aburatani, Hiroyuki Mano, Satoru Miyano, Seishi Ogawa, Yoshiyuki Takahashi, Hideki Muramatsu

    Blood   Vol. 131 ( 14 ) page: 1576 - 1586   2018.4

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    Juvenile myelomonocytic leukemia (JMML), a rare and aggressive myelodysplastic/myeloproliferative neoplasm that occurs in infants and during early childhood, is characterized by excessive myelomonocytic cell proliferation. More than 80% of patients harbor germ line and somatic mutations in RAS pathway genes (eg, PTPN11, NF1, NRAS, KRAS, and CBL), and previous studies have identified several biomarkers associated with poor prognosis. However, the molecular pathogenesis of 10% to 20% of patients and the relationships among these biomarkers have not been well defined. To address these issues, we performed an integrated molecular analysis of samples from 150 JMML patients. RNA-sequencing identified ALK/ROS1 tyrosine kinase fusions (DCTN1-ALK, RANBP2-ALK, and TBL1XR1-ROS1) in 3 of 16 patients (18%) who lacked canonical RAS pathway mutations. Crizotinib, an ALK/ROS1 inhibitor, markedly suppressed ALK/ROS1 fusion-positive JMML cell proliferation in vitro. Therefore, we administered crizotinib to a chemotherapy-resistant patient with the RANBP2-ALK fusion who subsequently achieved complete molecular remission. In addition, crizotinib also suppressed proliferation of JMML cells with canonical RAS pathway mutations. Genome-wide methylation analysis identified a hypermethylation profile resembling that of acute myeloid leukemia (AML), which correlated significantly with genetic markers with poor outcomes such as PTPN11/NF1 gene mutations, 2 or more genetic mutations, an AML-type expression profile, and LIN28B expression. In summary, we identified recurrent activated ALK/ROS1 fusions in JMML patients without canonical RAS pathway gene mutations and revealed the relationships among biomarkers for JMML. Crizotinib is a promising candidate drug for the treatment of JMML, particularly in patients with ALK/ROS1 fusions.

    DOI: 10.1182/blood-2017-07-798157

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  463. Phase I study of cord blood transplantation with intrabone marrow injection of mesenchymal stem cells: A clinical study protocol. Reviewed International journal

    Tatsunori Goto, Makoto Murata, Seitaro Terakura, Tetsuya Nishida, Yoshiya Adachi, Yoko Ushijima, Kazuyuki Shimada, Yuichi Ishikawa, Fumihiko Hayakawa, Nobuhiro Nishio, Satoshi Nishiwaki, Akihiro Hirakawa, Katsuyoshi Kato, Yoshiyuki Takahashi, Hitoshi Kiyoi

    Medicine   Vol. 97 ( 17 ) page: e0449   2018.4

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    INTRODUCTION: Delayed hematological recovery, graft failure, and acute graft-versus-host disease (GVHD) still remain major problems in cord blood transplantation (CBT). Mesenchymal stem cells (MSCs) are known to support bone marrow stroma and promote hematopoiesis. Additionally, MSCs possess immunomodulatory properties and are used clinically for the treatment of acute GVHD. Therefore, the use of MSCs to enhance engraftment and prevent GVHD after allogeneic hematopoietic cell transplantation has been explored. Recent clinical trials have shown the feasibility and safety of intravenous cotransplantation of MSCs with cord blood cells in pediatric patients, but not in adult patients, who are at greater risk of graft failure. As for the route of administration of MSCs, direct intrabone marrow injection of MSCs is thought to enhance the engraftment of cord blood cells more than intravenous injection. Based on these background findings, this clinical trial was designed to develop a new strategy to enhance engraftment and prevent GVHD after CBT. METHODS AND ANALYSIS: This is a single-center, phase I, clinical study to evaluate the safety of CBT combined with intrabone marrow injection of ex vivo expanded MSCs from bone marrow of a third-party donor. Adult patients with hematological disorders are eligible for this study. The target sample size is 5, and the registration period is 3 years. The target dose of MSCs infused is 0.5 × 10 cells/kg of patient body weight. On the day of CBT, MSCs are injected into the intrabone marrow of the patient 4 hours before the infusion of a single cord blood unit. The conditioning regimen varies according to patient age and disease. GVHD prophylaxis consists of a combination of tacrolimus and methotrexate. The primary endpoint of this study is infusional toxicity of MSCs within 14 days after transplantation.

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  464. Enhanced Expression of Anti-CD19 Chimeric Antigen Receptor in piggyBac Transposon-Engineered T Cells. Reviewed International journal

    Daisuke Morita, Nobuhiro Nishio, Shoji Saito, Miyuki Tanaka, Nozomu Kawashima, Yusuke Okuno, Satoshi Suzuki, Kazuyuki Matsuda, Yasuhiro Maeda, Matthew H Wilson, Gianpietro Dotti, Cliona M Rooney, Yoshiyuki Takahashi, Yozo Nakazawa

    Molecular therapy. Methods & clinical development   Vol. 8   page: 131 - 140   2018.3

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    Adoptive T cell therapy using chimeric antigen receptor (CAR)-modified T cells is a promising cancer immunotherapy. We previously developed a non-viral method of gene transfer into T cells using a piggyBac transposon system to improve the cost-effectiveness of CAR-T cell therapy. Here, we have further improved our technology by a novel culture strategy to increase the transfection efficiency and to reduce the time of T cell manufacturing. Using a CH2CH3-free CD19-specific CAR transposon vector and combining irradiated activated T cells (ATCs) as feeder cells and virus-specific T cell receptor (TCR) stimulation, we achieved 51.4% ± 14% CAR+ T cells and 2.8-fold expansion after 14 culture days. Expanded CD19.CAR-T cells maintained a significant fraction of CD45RA+CCR7+ T cells and demonstrated potent antitumor activity against CD19+ leukemic cells both in vitro and in vivo. Therefore, piggyBac-based gene transfer may provide an alternative to viral gene transfer for CAR-T cell therapy.

    DOI: 10.1016/j.omtm.2017.12.003

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  465. Comprehensive detection of pathogens in immunocompromised children with bloodstream infections by next-generation sequencing. Reviewed International journal

    Kazuhiro Horiba, Jun-Ichi Kawada, Yusuke Okuno, Nobuyuki Tetsuka, Takako Suzuki, Shotaro Ando, Yasuko Kamiya, Yuka Torii, Tetsuya Yagi, Yoshiyuki Takahashi, Yoshinori Ito

    Scientific reports   Vol. 8 ( 1 ) page: 3784 - 3784   2018.2

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    Bloodstream infection (BSI) is a severe complication in immunocompromised patients. Next-generation sequencing (NGS) allows us to analyze comprehensively and quantitatively all microorganisms present in a clinical sample. Thirty-five pediatric patients (12 with BSI and 23 with suspected BSI/negative blood culture) were enrolled. Plasma/serum samples were used for sequencing and the results were compared with those from blood culture. Sequencing reads of bacteria isolated in blood culture were identified by NGS in all plasma/serum samples at disease onset. Bacteria isolated in blood culture were identical to the dominant bacteria by NGS in 8 of 12 patients. Bacterial reads per million reads of the sequence depth (BR) > 200 and relative importance values of the dominant bacteria (P1) > 0.5 were employed to determine causative pathogens. Causative pathogens were detected using these criteria in 7 of 12 patients with BSI. Additionally, causative bacteria were detected in the plasma/serum at 7 days before disease onset in two patients with catheter-related BSI. Causative pathogens, including virus, were identified in three patients with suspected BSI. Lastly, a total of 62 resistance genes were detected by NGS. In conclusion, NGS is a new method to identify causative microorganisms in BSI and may predict BSI in some patients.

    DOI: 10.1038/s41598-018-22133-y

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  466. Comprehensive genetic analysis of donor cell derived leukemia with KMT2A rearrangement. Reviewed International journal

    Rieko Taniguchi, Hideki Muramatsu, Yusuke Okuno, Kyogo Suzuki, Satoshi Obu, Masahiro Nakatochi, Teppei Shimamura, Yoshiyuki Takahashi, Yasuo Horikoshi, Kenichiro Watanabe, Seiji Kojima

    Pediatric blood & cancer   Vol. 65 ( 2 )   2018.2

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    BACKGROUND: Donor cell leukemia (DCL) occurs after allogeneic hematopoietic stem cell transplantation. Several mechanisms, including occult leukemic/preleukemic subclones in the donor graft and germline predisposition to leukemia, are proposed to be associated with DCL's molecular pathogenesis. We report a comprehensive genetic analysis of a patient with KMT2A-rearranged DCL after allogeneic bone marrow transplantation for refractory cytopenia of childhood. PROCEDURE: We performed a whole-exome sequencing of the recipient's peripheral blood before transplant and the donor's peripheral blood and the recipient's bone marrow at the time of DCL diagnosis. RNA sequencing was also performed to detect fusion genes in DCL blasts. RESULTS: There were no germline mutations that were associated with a predisposition to leukemia in the recipient and donor. Furthermore, there were no detectable somatic alterations except KMT2A-MLLT10 and other related gene fusions in DCL. KMT2A-MLLT10 was not detectable in the donor's bone marrow. CONCLUSION: We propose a novel pattern of the molecular pathogenesis of DCL solely involving a genetic mutation acquired after transplant with no identifiable genetic factor related to the donor and recipient.

    DOI: 10.1002/pbc.26823

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  467. [5. Evaluation of Pathology of Heart Disease Models Using Preclinical Ultra-high Field MRI]. Reviewed

    Takahashi Y, Saito S

    Nihon Hoshasen Gijutsu Gakkai zasshi   Vol. 74 ( 4 ) page: 404 - 411   2018

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    DOI: 10.6009/jjrt.2018_JSRT_74.4.404

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  468. Neurocan, an extracellular chondroitin sulfate proteoglycan, stimulates neuroblastoma cells to promote malignant phenotypes. Reviewed International journal

    Zhendong Su, Satoshi Kishida, Shoma Tsubota, Kazuma Sakamoto, Dongliang Cao, Shinichi Kiyonari, Miki Ohira, Takehiko Kamijo, Atsushi Narita, Yinyan Xu, Yoshiyuki Takahashi, Kenji Kadomatsu

    Oncotarget   Vol. 8 ( 63 ) page: 106296 - 106310   2017.12

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    Neurocan (NCAN), a secreted chondroitin sulfate proteoglycan, is one of the major inhibitory molecules for axon regeneration in nervous injury. However, its role in cancer is not clear. Here we observed that high NCAN expression was closely associated with the unfavorable outcome of neuroblastoma (NB). NCAN was also highly and ubiquitously expressed in the early lesions and terminal tumor of TH-MYCN mice, a NB model. Interestingly, exogenous NCAN (i.e., overexpression, recombinant protein and conditioned medium) transformed adherent NB cells into spheres whose malignancies in vitro (anchorage-independent growth and chemoresistance) and in vivo (xenograft tumor growth) were potentiated. Both chondroitin sulfate sugar chains and NCAN's core protein were essential for the sphere formation. The CSG3 domain was essential in the moiety of NCAN. Our comprehensive microarray analysis and RT-qPCR of mRNA expression suggested that NCAN treatment promoted cell division, and urged cells to undifferentiated state. The knockdown of NCAN in tumor sphere cells cultured from TH-MYCN mice resulted in growth suppression in vitro and in vivo. Our findings suggest that NCAN, which stimulates NB cells to promote malignant phenotypes, is an extracellular molecule providing a growth advantage to cancer cells.

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  469. Bevacizumab, Irinotecan, and Temozolomide Combination as Salvage Chemotherapy for Relapsed Pediatric Brain Tumor

    Shinsuke Kataoka, Hideki Muramatsu, Kotaro Narita, Motoharu Hamada, Norihiro Murakami, Rieko Tanigushi, Daisuke Ichikawa, Hironori Kitazawa, Daiei Kojima, Kyogo Suzuki, Nozomu Kawashima, Eri Nishikawa, Atsushi Narita, Yusuke Okuno, Nobuhiro Nishio, Asahito Hama, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S55 - S55   2017.11

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  470. Comprehensive detection of viruses in pediatric patients with acute liver failure using next-generation sequencing. Reviewed International journal

    Takako Suzuki, Jun-Ichi Kawada, Yusuke Okuno, Satoshi Hayano, Kazuhiro Horiba, Yuka Torii, Yoshiyuki Takahashi, Syuichiro Umetsu, Tsuyoshi Sogo, Ayano Inui, Yoshinori Ito

    Journal of clinical virology : the official publication of the Pan American Society for Clinical Virology   Vol. 96   page: 67 - 72   2017.11

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    BACKGROUND: Pediatric acute liver failure (PALF) is a rare and severe syndrome that frequently requires liver transplantation. Viruses are one of the most frequent causes of this disease, however, pathogenic viruses are not determined in many patients. Recently next-generation sequencing (NGS) has been applied to comprehensively detect pathogens of infectious diseases of unknown etiology. OBJECTIVES: To evaluate an NGS-based approach for detecting pathogenic viruses in patients with PALF or acute hepatitis of unknown etiology. STUDY DESIGN: To detect virus-derived DNA and RNA sequences existing in sera/plasma from patients, both DNA and RNA sequencing were performed. First, we validated the ability of NGS to detect viral pathogens in clinical serum/plasma samples, and compared different commercial RNA library preparation methods Then, serum/plasma of fourteen patients with PALF or acute hepatitis of unknown etiology were evaluated using NGS. RESULTS: Among three RNA library preparation methods, Ovation RNA-Seq System V2 had the highest sensitivity to detect RNA viral sequences. Among fourteen patients, sequence reads of torque teno virus, adeno-associated virus, and stealth virus were found in the sera of one patient each, however, the pathophysiological role of these three viruses was not clarified. Significant virus reads were not detected in the remaining 11 patients. CONCLUSIONS: This finding might be due to low virus titer in blood at the time of referral or a non-infectious cause might be more frequent. These results suggest an NGS-based approach has potential to detect viral pathogens in clinical samples and would contribute to clarification of the etiology of PALF.

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  471. Clinical Trial of CD19 CAR-T Cell Therapy with non-virus Vector System

    Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S3 - S3   2017.11

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  472. Clinical Prognostic Factors in Patients with High-risk Neuroblastoma Undergoing A Single Autologous Stem Cell Transplant

    Yuya Saito, Chikako Kiyotani, Masami Inoue, Yoshiyuki Takahashi, Atsushi Ogawa, Motoaki Chin, Yuuki Koga, Koji Kato, Katsuyoshi Koh, Kenichiro Watanabe, Yoshiko Hashii, Atsushi Sato, Yuko Atsuta, Kimikazu Matsumoto

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S42 - S42   2017.11

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  473. Human Herpesvirus 6B Infection in Pediatric Hematopoietic Stem Cell Transplantation

    Hiroki Miura, Kazuko Kudo, Makito Tanaka, Yoshiyuki Takahashi, Seji Kojima, Koji Kato, Nao Yoshida, Hirotoshi Sakaguchi, Tetsushi Yoshikawa

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S76 - S76   2017.11

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  474. Identification of FLVCR1 as a Novel Causative Gene of Inherited Ineffective Erythropoiesis in Humans

    Nozomu Kawashima, Yusuke Okuno, Mitsutaka Shiota, Hideki Muramatsu, Kenichi Yoshida, Masahito Ikawa, Asahito Hama, Masafumi Ito, Sayoko Doisaki, Norihiro Murakami, Kyogo Suzuki, Atsushi Narita, Nobuhiro Nishio, Yoshiyuki Takahashi, Seishi Ogawa, Seiji Kojima

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S20 - S20   2017.11

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  475. Integrated Molecular Profiling of Juvenile Myelomonocytic Leukemia

    Norihiro Murakami, Hideki Muramatsu, Yusuke Okuno, Kenichi Yoshida, Yuichi Shiraishi, Genta Nagae, Kyogo Suzuki, Atsushi Narita, Hirotoshi Sakaguchi, Nozomu Kawashima, Xinan Wang, Yinyan Xu, Kenichi Chiba, Hiroko Tanaka, Nobuhiro Nishio, Asahito Hama, Masashi Sanada, Masashi Hirayama, Arata Watanabe, Toshihide Ueno, Seiji Kojima, Hiroyuki Aburatani, Hiroyuki Mano, Satoru Miyano, Seishi Ogawa, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S19 - S20   2017.11

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  476. Correlation of Bone Marrow Morphology with Clinical Findings and Gene Alterations in Patients with Diamond-Blackfan Anemia

    Asahito Hama, Tsutomu Toki, Akie Kobayashi, Hideki Muramatsu, Yusuke Okuno, Daisuke Hasegawa, Kazue Nozawa, Yoshiyuki Takahashi, Kenichiro Watanabe, Atsushi Manabe, Masafumi Ito, Etsuro Ito, Seiji Kojima

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S28 - S28   2017.11

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  477. Killer Cell Immunoglobulin-like Receptor Ligand-mismatched Allogeneic Cord Blood Transplantation Reduces Relapse and Improves Survival in Children with Refractory Neuroblastoma

    Nobuhiro Nishio, Shinnsuke Kataoka, Motoharu Hamada, Daisuke Ichikawa, Rieko Taniguchi, Norihiro Murakami, Daiei Kojima, Kyogo Suzuki, Nozomu Kawasima, Eri Nishikawa, Yusuke Okuno, Atsushi Narita, Hideki Muramatsu, Asahito Hama, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S23 - S24   2017.11

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  478. Germline Mutation in Pediatric Patients with Aplastic Anemia

    Atsushi Narita, Hideki Muramatsu, Yusuke Okuno, Xinan Wang, Yinyan Xu, Nozomu Kawashima, Nobuhiro Nishio, Asahito Hama, Hirotoshi Sakaguchi, Nao Yoshida, Koji Kato, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S28 - S29   2017.11

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  479. Vorinostat and Bortezomib Combined Intensive Chemotherapy for Children with <i>KMT2A</i> (<i>MLL</i>)-rearranged Acute Lymphoblastic Leukemia

    Suzuki, K; Muramatsu, H; Okuno, Y; Narita, K; Kitazawa, H; Huang, Y; Ichikawa, D; Kataoka, S; Hamada, M; Murakami, N; Taniguchi, R; Kojima, D; Sekiya, Y; Nishikawa, E; Kawashima, N; Narita, A; Nishio, N; Hama, A; Kojima, S; Takahashi, Y

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S90 - S90   2017.11

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  480. Newborn Screening for Severe Combined Immunodeficiency in Aichi Prefecture

    Daiei Kojima, Hideki Muramatsu, Yusuke Okuno, Kotaro Narita, Norihiro Murakami, Daisuke Ichikawa, Motoharu Hamada, Shinsuke Kataoka, Hironobu Kitazawa, Rieko Taniguchi, Kyogo Suzuki, Nozomu Kawashima, Eri Nishikawa, Atsushi Narita, Nobuhiro Nishio, Asahito Hama, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S33 - S33   2017.11

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  481. Retrospective Analysis of High Risk Childhood AML Undergoing Allo-SCT in First CR: Results of AML-05 by JPLSG

    Nobuyuki Hyakuna, Yoshiko Hashii, Hiroyuki Ishida, Katsutsugu Umeda, Yoshiyuki Takahashi, Masayuki Nagasawa, Hiromasa Yabe, Yozo Nakazawa, Katsuyoshi Koh, Hiroaki Goto, Hiroyuki Fujisaki, Kimikazu Matsumoto, Harumi Kakuda, Michihiro Yano, Akio Tawa, Daisuke Tomizawa, Takashi Taga, Souichi Adachi, Koji Kato

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S68 - S68   2017.11

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  482. Successful Allogeneic Cord Blood Transplantation in three Children with Aplastic Anemia Pretreated with Flu plus Mel plus TBI

    Eri Nishikawa, Hironobu Kitazawa, Koutarou Narita, Motoharu Hamada, Shinsuke Kataoka, Norihiro Murakami, Daisuke Ichikawa, Daiei Kojima, Kyogo Suzuki, Rieko Taniguchi, Nozomu Kawashima, Atsushi Narita, Yusuke Okuno, Hideki Muramatsu, Nobuhiro Nishio, Asahito Hama, Seiji Kojima, Yoshiyuki Takahashi

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S75 - S76   2017.11

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  483. Treatment Outcomes and Prognostic Factors after Allogeneic-HSCT for Patients with Low and Intermediate Risk in JPLSG AML-05 study

    Yoshiko Hashii, Nobuyuki Hyakuna, Hiroyuki Ishida, Katsutsugu Umeda, Yoshiyuki Takahashi, Masayuki Nagasawa, Hiromasa Yabe, Yozo Nakazawa, Hiroyuki Fujisaki, Michihiro Yano, Kimikazu Matsumoto, Hiroaki Goto, Harumi Kakuda, Katsuyoshi Koh, Daisuke Tomizawa, Akio Tawa, Takashi Taga, Souichi Adachi, Koji Kato

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S19 - S19   2017.11

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  484. Vorinostat and Bortezomib Combined Intensive Chemotherapy for Children with KMT2A (MLL)-rearranged Acute Lymphoblastic Leukemia

    Suzuki Kyogo, Muramatsu Hideki, Okuno Yusuke, Narita Kotaro, Kitazawa Hironobu, Huang Yue, Ichikawa Daisuke, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Taniguchi Rieko, Kojima Daiei, Sekiya Yuko, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Hama Asahito, Kojima Seiji, Takahashi Yoshiyuki

    PEDIATRIC BLOOD & CANCER   Vol. 64   page: S90-S90   2017.11

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  485. 難治性進行神経芽腫に対するKIRリガンド不一致同種臍帯血移植による再発抑制効果と生存率の向上(Killer cell immunoglobulin-like receptor ligand-mismatched allogeneic cord blood transplantation reduces relapse and improves survival in children with refractory neuroblastoma)

    Nishio Nobuhiro, Kataoka Shinsuke, Hamada Motoharu, Ichikawa Daisuke, Taniguchi Rieko, Murakami Norihiro, Kojima Daiei, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Okuno Yusuke, Narita Atsushi, Muramatsu Hideki, Hama Asahito, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 227 - 227   2017.10

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  486. 若年性骨髄単球性白血病に対する包括的遺伝子解析(Integrated molecular profiling of juvenile myelomonocytic leukemia)

    Murakami Norihiro, Muramatsu Hideki, Okuno Yusuke, Yoshida Kenichi, Shiraishi Yuichi, Nagae Genta, Suzuki Kyogo, Narita Atsushi, Sakaguchi Hirotoshi, Kawashima Nozomu, Wang Xinan, Xu Yinyan, Chiba Kenichi, Tanaka Hiroko, Nishio Nobuhiro, Hama Asahito, Sanada Masashi, Hirayama Masashi, Watanabe Arata, Ueno Toshihide, Kojima Seiji, Aburatani Hiroyuki, Mano Hiroyuki, Miyano Satoru, Ogawa Seishi, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 224 - 224   2017.10

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  487. 愛知県における重症複合免疫不全症に対する新生児マススクリーニング(Newborn screening for severe combined immunodeficiency in Aichi Prefecture)

    Kojima Daiei, Muramatsu Hideki, Okuno Yusuke, Narita Kotaro, Murakami Norihiro, Ichikawa Daisuke, Hamada Motoharu, Kataoka Shinsuke, Kitazawa Hironobu, Taniguchi Rieko, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Narita Atsushi, Nishio Nobuhiro, Hama Asahito, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 238 - 238   2017.10

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  488. 小児再生不良性貧血における胚細胞変異(Germline mutation in pediatric patients with aplastic anemia)

    Narita Atsushi, Muramatsu Hideki, Okuno Yusuke, Wang Xinan, Xu Yinyan, Kawashima Nozomu, Nishio Nobuhiro, Hama Asahito, Sakaguchi Hirotoshi, Yoshida Nao, Kato Koji, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 233 - 233   2017.10

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  489. 再発小児脳腫瘍に対するベバシズマブ・イリノテカン・テモゾロミド併用救援化学療法(Bevacizumab, Irinotecan, and Temozolomide combination as salvage chemotherapy for relapsed pediatric brain tumor)

    Kataoka Shinsuke, Muramatsu Hideki, Narita Kotaro, Hamada Motoharu, Murakami Norihiro, Tanigushi Rieko, Ichikawa Daisuke, Kitazawa Hironori, Kojima Daiei, Suzuki Kyogo, Kawashima Nozomu, Nishikawa Eri, Narita Atsushi, Okuno Yusuke, Nishio Nobuhiro, Hama Asahito, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 256 - 256   2017.10

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  490. ヒト遺伝性無効造血の原因となる新規FLVCR1遺伝子変異の同定(Identification of FLVCR1 as a novel causative gene of inherited ineffective erythropoiesis in humans)

    Kawashima Nozomu, Okuno Yusuke, Shiota Mitsutaka, Muramatsu Hideki, Yoshida Kenichi, Ikawa Masahito, Hama Asahito, Ito Masafumi, Doisaki Sayoko, Murakami Norihiro, Suzuki Kyogo, Narita Atsushi, Nishio Nobuhiro, Takahashi Yoshiyuki, Ogawa Seishi, Kojima Seiji

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 224 - 224   2017.10

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  491. KMT2A(MLL)遺伝子再構成陽性急性リンパ性白血病に対するボリノスタット・ボルテゾミブ併用化学療法(Vorinostat and bortezomib combined intensive chemotherapy for children with KMT2A(MLL)-rearranged acute lymphoblastic leukemia)

    Suzuki Kyogo, Muramatsu Hideki, Okuno Yusuke, Narita Kotaro, Kitazawa Hironobu, Huang Yue, Ichikawa Daisuke, Kataoka Shinsuke, Hamada Motoharu, Murakami Norihiro, Taniguchi Rieko, Kojima Daiei, Sekiya Yuko, Nishikawa Eri, Kawashima Nozomu, Narita Atsushi, Nishio Nobuhiro, Hama Asahito, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 287 - 287   2017.10

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  492. Flu+Mel+TBI前処置で同種臍帯血移植を施行した小児再生不良性貧血の3例(Successful allogeneic cord blood transplantation in three children with aplastic anemia pretreated with Flu+Mel+TBI)

    Nishikawa Eri, Kitazawa Hironobu, Narita Kotaro, Hamada Motoharu, Kataoka Shinsuke, Murakami Norihiro, Ichikawa Daisuke, Kojima Daiei, Suzuki Kyogo, Taniguchi Rieko, Kawashima Nozomu, Narita Atsushi, Okuno Yusuke, Muramatsu Hideki, Nishio Nobuhiro, Hama Asahito, Kojima Seiji, Takahashi Yoshiyuki

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 274 - 274   2017.10

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  493. Development of clinical paroxysmal nocturnal haemoglobinuria in children with aplastic anaemia. Reviewed International journal

    Atsushi Narita, Hideki Muramatsu, Yusuke Okuno, Yuko Sekiya, Kyogo Suzuki, Motoharu Hamada, Shinsuke Kataoka, Daisuke Ichikawa, Rieko Taniguchi, Norihiro Murakami, Daiei Kojima, Eri Nishikawa, Nozomu Kawashima, Nobuhiro Nishio, Asahito Hama, Yoshiyuki Takahashi, Seiji Kojima

    British journal of haematology   Vol. 178 ( 6 ) page: 954 - 958   2017.9

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    The clinical significance of paroxysmal nocturnal haemoglobinuria (PNH) in children with aplastic anaemia (AA) remains unclear. We retrospectively studied 57 children with AA between 1992 and 2010. During the follow-up, five patients developed clinical PNH, in whom somatic PIGA mutations were detected by targeted sequencing. The 10-year probability of clinical PNH development was 10·2% (95% confidence interval, 3·6-20·7%). Furthermore, the detection of minor PNH clones by flow cytometry at AA diagnosis was a risk factor for the subsequent development of clinical PNH. These patients with PNH clones at AA diagnosis should undergo periodic monitoring for potential clinical PNH development.

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  494. 若年性骨髄単球性白血病に対する包括的遺伝子解析(Integrated molecular profiling of juvenile myelomonocytic leukemia)

    Murakami Norihiro, Okuno Yusuke, Yoshida Kenichi, Shiraishi Yuichi, Nagae Genta, Suzuki Kyogo, Narita Atsushi, Sakaguchi Hirotoshi, Kawashima Nozomu, Wang Xinan, Xu Yinyan, Chiba Kenichi, Tanaka Hiroko, Hama Asahito, Sanada Masashi, Hirayama Masashi, Watanabe Arata, Ueno Toshihide, Kojima Seiji, Aburatani Hiroyuki, Mano Hiroyuki, Miyano Satoru, Ogawa Seishi, Takahashi Yoshiyuki, Muramatsu Hideki

    臨床血液   Vol. 58 ( 9 ) page: 1457 - 1457   2017.9

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  495. 先天性および後天性造血不全375例に対するクリニカルシーケンス(Clinical sequencing of 375 patients with inherited and acquired bone marrow failure syndromes)

    Muramatsu Hideki, Okuno Yusuke, Yoshida Kenichi, Shiraishi Yuichi, Narita Atsushi, Sakaguchi Hirotoshi, Kawashima Nozomu, Xu Yinyan, Chiba Kenichi, Tanaka Hiroko, Hama Asahito, Sanada Masashi, Takahashi Yoshiyuki, Kanno Hitoshi, Yamaguchi Hiroki, Ohga Shouichi, Manabe Atsushi, Harigae Hideo, Kunishima Shinji, Ishii Eiichi, Kobayashi Masao, Koike Kenichi, Watanabe Kenichiro, Ito Etsuro, Takata Minoru, Yabe Miharu, Miyano Satoru, Ogawa Seishi, Kojima Seiji

    臨床血液   Vol. 58 ( 9 ) page: 1519 - 1519   2017.9

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  496. Comparison of sperm subpopulation structures in first and second ejaculated semen from Japanese black bulls by a cluster analysis of sperm motility evaluated by a CASA system Reviewed

    Chihiro Kanno, Kentaro Q. Sakamoto, Yojiro Yanagawa, Yoshiyuki Takahashi, Seiji Katagiri, Masashi Nagano

    JOURNAL OF VETERINARY MEDICAL SCIENCE   Vol. 79 ( 8 ) page: 1359 - 1365   2017.8

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    In the present study, bull sperm in the first and second ejaculates were divided into subpopulations based on their motility characteristics using a cluster analysis of data from computer-assisted sperm motility analysis (CASA). Semen samples were collected from 4 Japanese black bulls. Data from 9,228 motile sperm were classified into 4 clusters; 1) very rapid and progressively motile sperm, 2) rapid and circularly motile sperm with widely moving heads, 3) moderately motile sperm with heads moving frequently in a short length, and 4) poorly motile sperm. The percentage of cluster 1 varied between bulls. The first ejaculates had a higher proportion of cluster 2 and lower proportion of cluster 3 than the second ejaculates.

    DOI: 10.1292/jvms.17-0012

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  497. Bone marrow transplantation for children with acquired bone marrow failure

    Hama A., Manabe A., Hasegawa D., Nozawa K., Suzuki K., Narita A., Muramatsu H., Takahashi Y., Watanabe K., Ohara A., Ito M., Kojima S.

    BONE MARROW TRANSPLANTATION   Vol. 52   page: S414 - S414   2017.7

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  498. Clinical utility of next-generation sequencing for inherited bone marrow failure syndromes. Reviewed International journal

    Hideki Muramatsu, Yusuke Okuno, Kenichi Yoshida, Yuichi Shiraishi, Sayoko Doisaki, Atsushi Narita, Hirotoshi Sakaguchi, Nozomu Kawashima, Xinan Wang, Yinyan Xu, Kenichi Chiba, Hiroko Tanaka, Asahito Hama, Masashi Sanada, Yoshiyuki Takahashi, Hitoshi Kanno, Hiroki Yamaguchi, Shouichi Ohga, Atsushi Manabe, Hideo Harigae, Shinji Kunishima, Eiichi Ishii, Masao Kobayashi, Kenichi Koike, Kenichiro Watanabe, Etsuro Ito, Minoru Takata, Miharu Yabe, Seishi Ogawa, Satoru Miyano, Seiji Kojima

    Genetics in medicine : official journal of the American College of Medical Genetics   Vol. 19 ( 7 ) page: 796 - 802   2017.7

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    PURPOSE: Precise genetic diagnosis of inherited bone marrow failure syndromes (IBMFS), a heterogeneous group of genetic disorders, is challenging but essential for precise clinical decision making. METHODS: We analyzed 121 IBMFS patients using a targeted sequencing covering 184 associated genes and 250 IBMFS patients using whole-exome sequencing (WES). RESULTS: We achieved successful genetic diagnoses for 53 of 121 patients (44%) using targeted sequencing and for 68 of 250 patients (27%) using WES. In the majority of cases (targeted sequencing: 45/53, 85%; WES: 63/68, 93%), the detected variants were concordant with, and therefore supported, the clinical diagnoses. However, in the remaining 13 cases (8 patients by target sequencing and 5 patients by WES), the clinical diagnoses were incompatible with the detected variants. CONCLUSION: Our approach utilizing targeted sequencing and WES achieved satisfactory diagnostic rates and supported the efficacy of massive parallel sequencing as a diagnostic tool for IBMFS.Genet Med advance online publication 19 January 2017.

    DOI: 10.1038/gim.2016.197

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  499. PiggyBac transposon, a non-viral gene transfer system, mediated T cells expressing CD19 chimeric antigen receptor for a 'first in human' clinical trial

    Takahashi, Y; Nishio, N; Hamada, M; Tanaka, M; Morita, D; Kawashima, N; Okuno, Y; Narita, A; Muramatsu, H; Hama, A; Kojima, S; Dotti, G; Nakazawa, Y

    BONE MARROW TRANSPLANTATION   Vol. 52   page: S161 - S162   2017.7

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  500. Recurrent MYB rearrangement in blastic plasmacytoid dendritic cell neoplasm

    K Suzuki, Y Suzuki, A Hama, H Muramatsu, M Nakatochi, M Gunji, D Ichikawa, M Hamada, R Taniguchi, S Kataoka, N Murakami, D Kojima, Y Sekiya, E Nishikawa, N Kawashima, A Narita, N Nishio, Y Nakazawa, H Iwafuchi, K-i Watanabe, Y Takahashi, M Ito, S Kojima, S Kato, Y Okuno

    Leukemia   Vol. 31 ( 7 ) page: 1629 - 1633   2017.7

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    DOI: 10.1038/leu.2017.101

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    Other Link: http://www.nature.com/articles/leu2017101

  501. 肺高血圧症(PH)をきたしtadalafilで治療した造血幹細胞移植(HSCT)後の3例

    深澤 佳絵, 早野 聡, 沼口 敦, 高橋 義行, 加藤 太一

    日本小児循環器学会雑誌   Vol. 33 ( Suppl.1 ) page: s1 - 402   2017.7

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  502. PiggyBac transposon, a non-viral gene transfer system, mediated T cells expressing CD19 chimeric antigen receptor for a 'first in human' clinical trial

    Takahashi Y, Nishio N, Hamada M, Tanaka M, Morita D, Kawashima N, Okuno Y, Narita A, Muramatsu H, Hama A, Kojima S, Dotti G, Nakazawa Y

    BONE MARROW TRANSPLANTATION   Vol. 52   page: S161-S162   2017.7

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  503. RECURRENT MYB REARRANGEMENT IN BLASTIC PLASMACYTOID DENDRITIC CELL NEOPLASM

    K. Suzuki, Y. Okuno, Y. Suzuki, A. Hama, H. Muramatsu, M. Nakatochi, M. Gunji, D. Ichikawa, M. Hamada, R. Taniguchi, S. Kataoka, N. Murakami, D. Kojima, Y. Sekiya, E. Nishikawa, N. Kawashima, A. Narita, N. Nishio, Y. Nakazawa, H. Iwafuchi, K. Watanabe, M. Ito, S. Kojima, S. Kato, Y. Takahashi

    HAEMATOLOGICA   Vol. 102   page: 35 - 35   2017.6

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  504. Efficacy of neutrophil non-muscle myosin heavy chain-IIA immunofluorescence analysis in determining the pathogenicity of MYH9 variants. Reviewed International journal

    Shinji Kunishima, Okuno Yusuke, Hideki Muramatsu, Daiei Kojima, Noriko Nagai, Yoshiyuki Takahashi, Seiji Kojima

    Annals of hematology   Vol. 96 ( 6 ) page: 1065 - 1066   2017.6

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    DOI: 10.1007/s00277-017-2972-3

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  505. Long-term outcomes of 95 children with moderate aplastic anemia treated with horse antithymocyte globulin and cyclosporine. Reviewed International journal

    Eri Nishikawa, Hiroshi Yagasaki, Asahito Hama, Hiromasa Yabe, Akira Ohara, Yoshiyuki Kosaka, Kazuko Kudo, Ryoji Kobayashi, Shouichi Ohga, Akira Morimoto, Ken-Ichiro Watanabe, Nao Yoshida, Hideki Muramatsu, Yoshiyuki Takahashi, Seiji Kojima

    Pediatric blood & cancer   Vol. 64 ( 5 )   2017.5

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    BACKGROUND: Currently, the standard management of moderate aplastic anemia (MAA) has not been well described, although the superiority of the combination of antithymocyte globulin (ATG) and cyclosporine (CyA) over CyA alone has been demonstrated in terms of hematological responses and failure-free survival (FFS). PROCEDURE: We adopted this therapeutic strategy and treated 95 children with MAA who were enrolled in two consecutive prospective studies between October 1992 and August 2009. RESULTS: For these patients, the 6-month response rate was 54.7% (complete response, 13.7%; partial response, 41.1%). There were no statistically significant differences in the overall response rates between the transfusion-dependent (48.8%, n = 41) and transfusion-independent groups (59.3%, n = 54; P = 0.4). Treatment failure was defined as the requirement of salvage treatment, and was observed in 52 patients. The 10-year FFS was 44.0% (95% confidence interval [CI], 32.9%-54.6%). Of the 22 patients who underwent a second immunosuppressive therapy (IST), 12 responded. Forty patients underwent hematopoietic stem cell transplantation as second- or third-line therapy and three died of complications. Consequently, the 10-year overall survival rate was 96.0% (95% CI, 88.0%-98.7%) with a median follow-up period of 103 months (range, 29-221 months). CONCLUSIONS: Although current guidelines recommend only observation for patients with transfusion-independent MAA, the results of our study justify early intervention with ATG and CyA in those patients. A prospective randomized trial is warranted to clarify the risks and benefits of early intervention with IST and observation alone until progression to severe AA in patients with MAA.

    DOI: 10.1002/pbc.26305

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  506. Paroxysmal nonepileptic events in children with epilepsy. Reviewed International journal

    Yuji Ito, Hiroyuki Kidokoro, Tamiko Negoro, Masaharu Tanaka, Yu Okai, Yoko Sakaguchi, Chikako Ogawa, Tomoya Takeuchi, Atsuko Ohno, Hiroyuki Yamamoto, Tomohiko Nakata, Satoshi Maesawa, Kazuyoshi Watanabe, Yoshiyuki Takahashi, Jun Natsume

    Epilepsy research   Vol. 132   page: 59 - 63   2017.5

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    OBJECTIVE: The aim of this study was to clarify the characteristics of paroxysmal nonepileptic events (PNEs) suspected as being epileptic seizures by families of children with epilepsy. METHODS: The video-EEG (vEEG) recordings of habitual paroxysmal events in children with epilepsy at Nagoya University Hospital between October 2006 and January 2016 were reviewed. Based on the doctor's suspicion before the vEEG, the PNEs were divided into two groups that included PNEs suspected as epileptic seizures and PNEs suspected as PNEs. PNEs in the former group were classified based on the suspected seizure type. RESULTS: Of 886 habitual paroxysmal events, vEEG confirmed that 83 events (68 children) were PNEs. The median age of the 68 children was 3.2 years. Concurrent epilepsies included focal epilepsies (n=33), infantile spasms (n=16), and other types (n=19). The most common types of PNEs were sleep myoclonus (n=11), followed by stereotypies (n=9), awake myoclonus (n=8), paroxysmal ocular deviations (PODs, n=8), and tonic posturing (n=8). Even after direct observation or video viewing, the doctors suspected epileptic seizures in all three of the PODs and two of the tonic posturing children. Before the vEEG, however, the accurate visual information led to the speculation that the four psychogenic and two sleep myoclonus events were all PNEs. Myoclonus, stereotypies, and head drops were often misdiagnosed as epileptic spasms, while PODs and tonic posturing were often misdiagnosed as focal seizures with motor components. Additionally, staring and motion arrest during a drowsy state were often misdiagnosed as focal dyscognitive seizures. Seven of eight patients with PODs had epileptic spasms that were concurrent with epileptic seizures. A diffuse cerebral lesion or reduced visual acuity was seen in seven patients with PODs. CONCLUSION: We re-emphasize that vEEG is essential for accurate diagnosis and provides evidence for listing POD in the differential diagnosis of oculomotor paroxysmal events.

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  507. PiggyBac Mediated T Cells Expressing Anti CD19 Chimeric Antigen Receptor for a Clinical Trial

    Nobuhiro Nishio, Yozo Nakazawa, Motoharu Hamada, Satoshi Suzuki, Miyuki Tanaka, Daisuke Morita, Nozomu Kawashima, Yusuke Okuno, Atsushi Narita, Asahito Hama, Hideki Muramatsu, Hideki Muramatsu, Seiji Kojima, Yoshiyuki Takahashi

    MOLECULAR THERAPY   Vol. 25 ( 5 ) page: 291 - 291   2017.5

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  508. Germline IKAROS mutation associated with primary immunodeficiency that progressed to T-cell acute lymphoblastic leukemia Reviewed

    N. Yoshida, H. Sakaguchi, H. Muramatsu, Y. Okuno, C. Song, S. Dovat, A. Shimada, M. Ozeki, H. Ohnishi, T. Teramoto, T. Fukao, N. Kondo, Y. Takahashi, K. Matsumoto, K. Kato, S. Kojima

    LEUKEMIA   Vol. 31 ( 5 ) page: 1221 - 1223   2017.5

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    DOI: 10.1038/leu.2017.25

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  509. Early germinal vesicle breakdown is a predictor of high preimplantation developmental competent oocytes in mice Reviewed

    Shogo Higaki, Masao Kishi, Keisuke Koyama, Masashi Nagano, Seiji Katagiri, Tatsuyuki Takada, Yoshiyuki Takahashi

    ZYGOTE   Vol. 25 ( 1 ) page: 41 - 48   2017.2

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    The preselection of highly developmentally competent oocytes for in vitro maturation (IVM) is crucial for improving assisted reproductive technology. Although several intrinsic markers of oocyte quality are known to be closely related to the onset of nuclear maturation (germinal vesicle break down, GVBD), a direct comparison between GVBD timing and oocyte quality has never been reported. In this study, we established a non-invasive oocyte evaluation method based on GVBD timing for preselecting more developmental competent oocytes in mice. Because the O-2 concentration during IVM may affect the nuclear kinetics, all experiments were performed under two distinct O-2 concentrations: 20% and 5% O-2. First, we determined the time course of changes in nuclear maturation and preimplantation developmental competence of in vitro-matured oocytes to estimate GVBD timing in high developmental competent oocytes. Two-thirds of oocytes that underwent GVBD in early IVM seemed to mainly contribute to the blastocyst yield. To confirm this result, we compared the preimplantation developmental competence of the early and late GVBD oocytes. Cleavage and blastocyst formation rates of early GVBD oocytes (80.2% and 52.7% under 20% O-2, respectively, and 67.6% and 47.3% under 5% O-2, respectively) were almost double those of late GVBD oocytes (44.8% and 26.0% under 20% O-2, respectively, and 40.4% and 17.9% under 5% O-2, respectively). With no observable alterations by checking the timing of GVBD in preimplantation developmental competence, oocyte evaluation based on GVBD timing can be used as an efficient and non-invasive preselection method for high developmental competent oocytes.

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  510. Investigation of a Rotavirus Gastroenteritis Outbreak among Immunosuppressed Patients in a Hospital Setting.

    Sugata K, Hull J, Wang H, Foytich K, Moon SS, Takahashi Y, Kojima S, Yoshikawa T, Jiang B

    Journal of immunological techniques in infectious diseases   Vol. 6 ( 1 )   2017.1

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    DOI: 10.4172/2329-9541.1000153

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  511. Clinical utility of next-generation sequencing-based minimal residual disease in paediatric B-cell acute lymphoblastic leukaemia. Reviewed International journal

    Yuko Sekiya, Yinyan Xu, Hideki Muramatsu, Yusuke Okuno, Atsushi Narita, Kyogo Suzuki, Xinan Wang, Nozomu Kawashima, Hirotoshi Sakaguchi, Nao Yoshida, Asahito Hama, Yoshiyuki Takahashi, Koji Kato, Seiji Kojima

    British journal of haematology   Vol. 176 ( 2 ) page: 248 - 257   2017.1

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    We assessed the clinical utility of next-generation sequencing (NGS)-based monitoring of minimal residual disease (MRD) in a uniformly treated cohort of 79 patients with paediatric B-cell acute lymphoblastic leukaemia. Bone marrow samples were collected at the time of diagnosis, days 33 and 80, pre- (4-5 months) and post- (24 months) maintenance therapy time points, and at relapse. We identified leukaemia-specific CDR3 sequences in 72 of 79 patients (91%) and detected MRD in 59 of 232 samples. Although MRD was detected in 28 of 55 samples (51%) on day 33, the frequencies of MRD detection decreased to 25% (16/65) at day 80, 19% (11/58) at 4-5 months and 7·4% (4/54) at 24 months. In a univariate analysis, positive MRD results on day 80 [relative risk (RR) 95% confidence interval (CI) = 7·438 (2·561-21·6), P < 0·001], at 4-5 months [RR (95% CI) = 10·24 (3·374-31·06), P < 0·001], and at 24 months [RR (95% CI) = 19·26 (4·974-74·59), P < 0·001] exhibited statistically significant associations with inferior leukaemia-free survival; this was confirmed using a Cox proportional hazard model. Our study suggests the promising potential of NGS-MRD for patients with B-cell ALL.

    DOI: 10.1111/bjh.14420

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  512. Allogeneic hematopoietic stem cell transplantation using ATG: prophylaxis and treatment. Reviewed

    Nishio N, Takahashi Y, Kojima S

    [Rinsho ketsueki] The Japanese journal of clinical hematology   Vol. 58 ( 10 ) page: 2104 - 2110   2017

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    DOI: 10.11406/rinketsu.58.2104

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  513. The Measurement of X-ray Fluoroscopy Input-output Characteristics in Angiography System. Reviewed

    Hagihara Y, Umehara T, Matsumoto K, Fujita T, Ikeuchi Y, Fujikawa K, Takahashi Y, Kotoura N

    Nihon Hoshasen Gijutsu Gakkai zasshi   Vol. 73 ( 2 ) page: 96 - 103   2017

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    DOI: 10.6009/jjrt.2017_JSRT_73.2.96

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  514. *Relapse of leukemia with loss of mismatched HLA due to uniparental disomy following haploidentical hematopoietic stem cell transplantation. Reviewed

    Villalobos IB, Takahashi Y, Akatsuka Y, Muramatsu H, Nishio N, Hama A, Yagasaki H, Saji H, Kato M, Ogawa S, Kojima S.

    Blood   Vol. Epub ahead of print   page: Epub ahead of print   2010

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  515. *Regression of human kidney cancer following allogeneic stem cell transplantation is associated with recognition of an HERV-E antigen by T cells. Reviewed

    Takahashi, Y., N. Harashima, S. Kajigaya, H. Yokoyama, E. Cherkasova, J.P. McCoy, K.I. Hanada, O. Mena, R. Kurlander, T. Abdul, R. Srinivasan, A. Lundqvist, E. Malinzak, N. Geller, M.I. Lerman, and R.W. Childs.

    The Journal of Clinical Investigation   Vol. 118   page: 1099-1109.   2008

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  516. *In vitro and in vivo evidence of PNH cell sensitivity to immune attack after nonmyeloablative allogeneic hematopoietic cell transplantation. Reviewed

    Takahashi Y, McCoy JP Jr, Carvallo C, Rivera C, Igarashi T, Srinivasan R, Young NS, and Childs RW

    Blood   Vol. 103   page: 1383-1390   2004

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    Authorship:Lead author   Language:English   Publishing type:Research paper (scientific journal)  

  517. *An Allogeneic-Based Immunotherapy For Renal Cell Carcinoma. Invited Reviewed

    Takahashi Y, Childs, R

    Clinical Cancer Research   Vol. 10 ( 18Pt2 ) page: 6353S-6359S   2004

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    Authorship:Lead author   Language:English  

▼display all

Books 1

  1. *Hematopoietic stem cell transplantation for solid tumors.

    Takahashi Y, Srinivasan R, and Childs R( Role: Joint author)

    Handbook of Stem Cells  2004 

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    Language:English

MISC 152

  1. A 7-year-old boy who responded to vedolizumab for steroid-refractory gut graft-versus-host disease

    西川英里, 村松秀城, 吉田太郎, 今屋雅之, 若松学, 三輪田俊介, 山森彩子, 成田幸太郎, 谷口理恵子, 濱田太立, 川島希, 成田敦, 奥野友介, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 43rd   2021

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  2. Development of clinical test of DNA methylation classification for patients with JMML

    村松秀城, 北澤宏展, 奥野友介, 村上典寛, 若松学, 濱田太立, 川島希, 成田敦, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 43rd   2021

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  3. 診断および治療に難渋している,脳を含む全身性多発転移腫瘤を有する先天性肉腫の乳児

    今屋雅之, 川島希, 山森彩子, 若松学, 吉田太郎, 三輪田俊介, 成田幸太郎, 北澤宏展, 谷口理恵子, 市川大輔, 濱田太立, 西川英里, 成田敦, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 高橋義行, 中村栄男, 孝橋賢一, 大喜多肇

    日本小児血液・がん学会雑誌(Web)   Vol. 58 ( 1 )   2021

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  4. Outcome of hematopoietic cell transplantation in children with Fanconi anemia

    成田敦, 津村悠介, 前村遼, 今屋雅之, 山森彩子, 吉田太郎, 若松学, 三輪田俊介, 成田幸太郎, 谷口理恵子, 濱田太立, 西川英里, 川島希, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 43rd   2021

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  5. がん遺伝子パネル検査でBRAF V600E変異を認めた副腎腫瘍の16歳女性

    佐治木大知, 村松秀城, 津村悠介, 前村遼, 今屋雅之, 山森彩子, 若松学, 吉田太郎, 三輪田俊介, 成田幸太郎, 市川大輔, 濱田太立, 谷口理恵子, 鈴木喬悟, 川島希, 西川英里, 成田敦, 西尾信博, 高橋義行, 島田聡子, 榊原綾子, 下山芳江, 岩淵英人

    日本小児血液・がん学会雑誌(Web)   Vol. 58 ( 3 )   2021

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  6. がん遺伝子パネル検査でGOPC-ROS1fusionを認めた中枢神経胎児性腫瘍の0歳児症例

    夏目敦至, 栗本路弘, 芝良樹, 青木恒介, 若林俊彦, 川島希, 谷口理恵子, 村松秀城, 高橋義行, 高野桂, 榊原綾子, 下山芳江, 中村栄男, 森田佐知, 安藤雄一, 平戸純子

    日本小児血液・がん学会雑誌(Web)   Vol. 58 ( 1 )   2021

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  7. A case of aggressive systemic mastocytosis who received R-BMT after nilotinib therapy

    前村遼, 村松秀城, 吉田太郎, 今屋雅之, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 谷口理恵子, 市川大輔, 濱田太立, 西川英里, 川島希, 成田敦, 奥野友介, 西尾信博, 小島勢二, 三浦浩樹, 田中真己人, 工藤寿子, 吉川哲史, 山田勢至, 伊藤雅文, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 43rd   2021

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  8. Efficacy of Prelixafor for stem cell mobilization in cihldren with relapse/refractory neuroblastoma

    濱田太立, 成田敦, 今屋雅之, 山森彩子, 吉田太郎, 若松学, 三輪田俊介, 成田幸太郎, 川島希, 谷口理恵子, 西川英里, 西尾信博, 西尾信博, 村松秀城, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 43rd   2021

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  9. Graft-versus-tumor effect of KIR ligand mismatched CBT for high-risk metastatic neuroblastoma

    西尾信博, 西尾信博, 市川大輔, 山森彩子, 若松学, 成田幸太郎, 吉田太郎, 今屋雅之, 三輪田俊介, 谷口理恵子, 濱田太立, 西川英里, 川島希, 奥野友介, 奥野友介, 成田敦, 村松秀城, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 43rd   2021

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  10. Diagnostic Whole Exome Sequencing for 166 Patients with Inherited Bone Marrow Failure Syndrome

    Motoharu Hamada, Hideki Muramatsu, Yusuke Okuno, Ayako Yamamori, Taro Yoshida, Masayuki Imaya, Manabu Wakamatsu, Shunsuke Miwata, Kotaro Narita, Daisuke Ichikawa, Rieko Taniguchi, Atsushi Narita, Nozomu Kawashima, Eri Nishikawa, Nobuhiro Nishio, Asahito Hama, Masafumi Ito, Tomoo Ogi, Seiji Kojima, Yoshiyuki Takahashi

    BLOOD   Vol. 136   2020.11

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    Language:English   Publishing type:Research paper, summary (international conference)   Publisher:AMER SOC HEMATOLOGY  

    0

    DOI: 10.1182/blood-2020-143241

    Web of Science

  11. KIR ligand mismatched CBT following consolidation chemotherapy for high risk metastatic neuroblastoma

    西尾信博, 西尾信博, 市川大輔, 山森彩子, 若松学, 成田幸太郎, 吉田太郎, 今屋雅之, 三輪田俊介, 谷口理恵子, 濱田太立, 西川英里, 川島希, 奥野友介, 奥野友介, 成田敦, 村松秀城, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  12. A case of SCID who caused in-hospital transmission of vaccine-derived rotavirus strain after transplantation

    成田幸太郎, 村松秀城, 吉田太郎, 山森彩子, 若松学, 三輪田俊介, 今屋雅之, 北澤宏展, 谷口理恵子, 市川大輔, 濱田太立, 西川英里, 西尾信博, 奥野友介, 川島希, 成田敦, 小島勢二, 安岡竜平, 三浦浩樹, 吉川哲史, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  13. 移植後ブリナツモマブ投与後に移植関連TMAを発症した急性リンパ性白血病の6歳女児

    今屋雅之, 村松秀城, 山森彩子, 若松学, 吉田太郎, 三輪田俊介, 成田幸太郎, 谷口理恵子, 濱田太立, 市川大輔, 西川英里, 川島希, 成田敦, 奥野友介, 西尾信博, 小島勢二, 高橋義行

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  14. 自己炎症疾患におけるプロテアソーム・サブユニットの異常が優性阻害によりプロテアソーム機能を喪失させる

    川島希, 片岡伸介, 村松秀城, 大西秀典, 奥野友介, 今屋雅之, 吉田太郎, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 濱田太立, 市川大輔, 西川英里, 成田敦, 西尾信博, 小島勢二, 高橋義行

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  15. A case of CBT for congenital leukemia with KAT6A-EP300 fusion gene that relapsed after spontaneous remission

    三輪田俊介, 谷口理恵子, 伊川泰広, 成田敦, 濱田太立, 川島希, 吉田太郎, 今屋雅之, 山森彩子, 若松学, 成田幸太郎, 北澤宏展, 市川大輔, 西川英里, 奥野友介, 村松秀城, 西尾信博, 西村良成, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  16. 自然寛解後に新たにMYB-GMDS融合遺伝子を獲得して再発した先天性急性骨髄性白血病

    谷口理恵子, 村松秀城, 伊川泰広, 濱田太立, 川島希, 吉田太郎, 今屋雅之, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 市川大輔, 西川英里, 成田敦, 奥野友介, 西尾信博, 西尾信博, 西村良成, 小島勢二, 高橋義行

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  17. 術前診断にて良性腫瘍と考えられたが全摘出できなかった腰髄腫瘍の2歳男児

    吉田太郎, 村松秀城, 今屋雅之, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 北澤宏展, 谷口理恵子, 市川大輔, 濱田太立, 西川英里, 川島希, 成田敦, 西尾信博, 小島勢二, 高橋義行, 田中智史, 安藤圭, 小林和克, 今釜史郎, 中黒匡人, 平戸純子

    日本小児血液・がん学会雑誌(Web)   Vol. 57 ( 5 )   2020

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  18. 遺伝性骨髄不全症候群166例に対する全エクソン解析

    濱田太立, 村松秀城, 奥野友介, 山森彩子, 吉田太郎, 今屋雅之, 若松学, 三輪田俊介, 成田幸太郎, 市川大輔, 谷口理恵子, 成田敦, 川島希, 西川英里, 西尾信博, 濱麻人, 伊藤雅文, 荻朋男, 小島勢二, 高橋義行

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  19. 重症再生不良性貧血における抗ヒト胸腺細胞ウサギ免疫グロブリンを用いた免疫抑制療法後のCMV/EBV再活性化

    成田敦, 伊藤嘉規, 村松秀城, 濱田太立, 市川大輔, 西川英里, 川島希, 奥野友介, 西尾信博, 鳥居ゆか, 川田潤一, 山崎宏人, 中尾眞二, 小島勢二, 高橋義行

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  20. 重症複合免疫不全症に対し前処置なし同種臍帯血移植で感染制御後に前処置あり同種臍帯血移植を実施した1例

    成田幸太郎, 村松秀城, 吉田太郎, 山森彩子, 若松学, 今屋雅之, 三輪田俊介, 谷口理恵子, 市川大輔, 濱田太立, 西川英里, 西尾信博, 奥野友介, 川島希, 成田敦, 高橋義行

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  21. Multiprofessional involvement in long-term follow-up of pediatric patients after HSCT

    西川栄里, 吉田太郎, 今屋雅之, 若松学, 山森彩子, 三輪田俊介, 成田幸太郎, 北澤宏展, 谷口理恵子, 濱田太立, 市川大輔, 川島希, 成田敦, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  22. Blinatumomab treatment prior to HCT for pediatric patients, a single-institution experience

    北澤宏展, 成田敦, 吉田太郎, 今屋雅之, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 谷口理恵子, 濱田太立, 市川大輔, 西川英里, 川島希, 奥野友介, 西尾信博, 村松秀城, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  23. Successful treatment using oral budesonide for gastrointestinal GVHD resistant to mesenchymal stem cell therapy

    吉田太郎, 西川英里, 今屋雅之, 若松学, 三輪田俊介, 成田幸太郎, 山森彩子, 北澤宏展, 谷口理恵子, 市川大輔, 濱田太立, 川島希, 成田敦, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  24. 化学療法による治療介入を行った生後2か月未満発症の神経芽腫4S

    今屋雅之, 西川英里, 吉田太郎, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 北澤宏展, 谷口理恵子, 市川大輔, 濱田太立, 川島希, 成田敦, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本小児血液・がん学会雑誌(Web)   Vol. 57 ( 5 )   2020

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  25. Neutropenia after allogeneic hematopoietic stem cell transplant

    川島希, 今屋雅之, 吉田太郎, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 北澤宏展, 濱田太立, 市川大輔, 西川英里, 奥野友介, 成田敦, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  26. Analysis of fulminant adenovirus hepatitis after hematopoietic stem cell transplantation in children

    市川大輔, 川島希, 吉田太郎, 今屋雅之, 若松学, 三輪田俊介, 成田幸太郎, 山森彩子, 北澤宏展, 谷口理恵子, 濱田太立, 西川英里, 成田敦, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 川田潤一, 伊藤嘉規, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  27. Comparison of EASIx and GFR predicting early transplantation-related complications in children undergoing hematopoietic stem cell transplant

    今屋雅之, 山森彩子, 若松学, 吉田太朗, 三輪田俊介, 成田幸太郎, 北澤宏展, 谷口理恵子, 市川大輔, 濱田太立, 西川英里, 川島希, 成田敦, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  28. Genome-wide methylation analysis of patients with juvenile myelomonocytic leukemia who received allogeneic stem cell transplantation

    村松秀城, 村上典寛, 奥野友介, 若松学, 北澤宏展, 濱田太立, 川島希, 成田敦, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 42nd   2020

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  29. CD19.CAR-T療法後の治療決定におけるNGS based MRDの有用性の検討

    西尾信博, 西尾信博, 濱田太立, 奥野友介, 奥野友介, 今屋雅之, 吉田太郎, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 市川大輔, 川島希, 西川英里, 成田敦, 村松秀城, WANG Kai, ZHENG Huyong, CHANG Lung-Ji, CHANG Lung-Ji, CHANG Lung-Ji, 小島勢二, 高橋義行

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  30. CD19CAR-T療法を行った再発MEF2D-BCL9陽性小児急性リンパ性白血病の1例

    吉田太郎, 村松秀城, 今屋雅之, 山森彩子, 若松学, 三輪田俊介, 成田幸太郎, 谷口理恵子, 市川大輔, 西川英里, 濱田太立, 成田敦, 川島希, 西尾信博, 奥野友介, 小島勢二, 高橋義行, 入江正寛, 笹原洋二

    日本血液学会学術集会抄録(Web)   Vol. 82nd   2020

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  31. 院内で急変した小児の蘇生状況に関する多施設共同研究

    大萱 俊介, 沼口 敦, 伊藤 祥絵, 田上 和憲, 阿部 ちひろ[羽田野], 宮地 悠江, 川田 潤一, 夏目 淳, 小島 勢二, 高橋 義行

    日本小児科学会雑誌   Vol. 123 ( 4 ) page: 734 - 740   2019.4

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    3年間に院内で急変した小児の蘇生状況を後方視的に調査した。調査対象36施設のうちIntensive Care Unit(ICU)併設施設は16施設(44%)であったが、Pediatric Intensive Care Unit併設施設はなく、21施設から計355例の重症患者が回答された。院内で急変した小児に対する蘇生処置について回答の得られた76例のうち、48例(63%)は一般病棟で急変しており、ICU管理中の発症は11例(14%)であった。全体の自己心拍再開(ROSC)率は55%、生存退院率は28%と生命予後は悪い傾向を示し、蘇生場所によるROSC率、生存退院率に有意差は認めなかった。また、中核病院での発生割合は14%であり、小児重症患者の中核病院への集約化が進んでいない現状が明らかとなった。

  32. 肋骨部腫瘤から脊椎浸潤をきたしOncologic emergencyで紹介された1歳男児

    三輪田 俊介, 市川 大輔, 成田 幸太郎, 北澤 宏展, 村上 典寛, 濱田 太立, 片岡 伸介, 谷口 理恵子, 鈴木 喬悟, 川島 希, 西川 英里, 成田 敦, 村松 秀城, 西尾 信博, 奥野 友介, 小島 勢二, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 56 ( 1 ) page: 69 - 69   2019.4

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  33. キイトルーダによる尋常性天疱瘡、再投与にて再燃を認めた1例

    水谷 育宏, 神野 義行, 高橋 直希, 黒田 直人

    西日本皮膚科   Vol. 81 ( 2 ) page: 134 - 134   2019.4

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  34. 東海地区IRUDにおける未診断疾患152家系のトリオ全エクソーム解析

    成田 幸太郎, 村松 秀城, 加藤 耕治, 片岡 伸介, 中村 勇治, 浅田 英之, 村松 友佳子, 城所 博之, 久保田 哲夫, 要 匡, 齋藤 伸治, 高橋 義行, 小島 勢二

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 282 - 282   2019.2

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  35. 末節骨短縮型点状軟骨異形成症と3C症候群の表現型を呈した姉弟例

    加藤 耕治, 河野 好彦, 澤村 文, 原 紳也, 村松 秀城, 高橋 義行, 齋藤 伸治

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 403 - 403   2019.2

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  36. 末節骨短縮型点状軟骨異形成症と3C症候群の表現型を呈した姉弟例

    加藤 耕治, 河野 好彦, 澤村 文, 原 紳也, 村松 秀城, 高橋 義行, 齋藤 伸治

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 403 - 403   2019.2

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  37. 東海地区IRUDにおける未診断疾患152家系のトリオ全エクソーム解析

    成田 幸太郎, 村松 秀城, 加藤 耕治, 片岡 伸介, 中村 勇治, 浅田 英之, 村松 友佳子, 城所 博之, 久保田 哲夫, 要 匡, 齋藤 伸治, 高橋 義行, 小島 勢二

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 282 - 282   2019.2

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  38. 当院の新生児集中治療室における敗血症の起炎微生物と初期治療薬の妥当性

    手塚 宜行, 早川 昌弘, 松本 あかね, 佐藤 義朗, 齊藤 明子, 村松 友佳子, 伊藤 美春, 鈴木 俊彦, 田中 亮, 北瀬 悠磨, 三浦 良介, 浅田 英之, 兵藤 玲奈, 脇田 浩正, 高橋 義行

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 338 - 338   2019.2

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  39. 小児血液・腫瘍患者における中心静脈カテーテルの計画外抜去リスクについての検討

    宮城島 萌, 濱田 太立, 村松 秀城, 奥野 友介, 鈴木 喬悟, 川島 希, 成田 敦, 西川 英里, 西尾 信博, 高橋 義行

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 266 - 266   2019.2

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  40. 先天性横隔膜ヘルニアにおける中期的神経学的予後と頭部MRI画像についての検討

    伊藤 美春, 脇田 浩正, 郷間 環, 上田 一仁, 田中 亮, 三浦 良介, 呉 尚治, 兵藤 玲奈, 浅田 英之, 北瀬 悠磨, 鈴木 俊彦, 見松 はるか, 齊藤 明子, 村松 友佳子, 城所 博之, 佐藤 義朗, 夏目 淳, 高橋 義行, 早川 昌弘

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 337 - 337   2019.2

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  41. 小児血液・腫瘍患者における中心静脈カテーテルの計画外抜去リスクについての検討

    宮城島 萌, 濱田 太立, 村松 秀城, 奥野 友介, 鈴木 喬悟, 川島 希, 成田 敦, 西川 英里, 西尾 信博, 高橋 義行

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 266 - 266   2019.2

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  42. CNS-ALLマウスモデルでのpiggyBac CD19 CAR-T細胞脳室内投与の安全性と有効性の検討(Direct delivery of piggyBac CD19 CAR-T cells against CNS-ALL xenograft model)

    田中 邦昭, 加藤 格, 田中 美幸, 盛田 大介, 松田 和之, 高橋 義行, 梅田 雄嗣, 平松 英文, 中畑 龍俊, 足立 壯一, 滝田 順子, 中沢 洋三

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 262 - 262   2019.2

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  43. CNS-ALLマウスモデルでのpiggyBac CD19 CAR-T細胞脳室内投与の安全性と有効性の検討(Direct delivery of piggyBac CD19 CAR-T cells against CNS-ALL xenograft model)

    田中 邦昭, 加藤 格, 田中 美幸, 盛田 大介, 松田 和之, 高橋 義行, 梅田 雄嗣, 平松 英文, 中畑 龍俊, 足立 壯一, 滝田 順子, 中沢 洋三

    日本小児科学会雑誌   Vol. 123 ( 2 ) page: 262 - 262   2019.2

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  44. 優秀演題賞候補 重症新生児仮死児におけるaEEGの新たな背景活動パターンとその臨床的意義

    田中 雅大, 城所 博之, 川口 将宏, 牧 祐輝, 岡井 佑, 坂口 陽子, 山本 啓之, 中田 智彦, 渡邊 一功, 高橋 義行, 夏目 淳, 久保田 哲夫, 深沢 達也, 根来 民子, 奥村 彰久, 加藤 徹

    日本小児科学会雑誌   Vol. 123 ( 1 ) page: 102 - 102   2019.1

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  45. 【がん免疫の最前線】キメラ抗原受容体遺伝子改変T細胞療法の最前線

    西尾 信博, 高橋 義行

    BIO Clinica   Vol. 34 ( 1 ) page: 20 - 24   2019.1

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    キメラ抗原受容体遺伝子改変T細胞(CAR-T)療法は、CD19陽性B細胞性血液悪性腫瘍に対して従来と比較して優れた臨床効果を発揮し、最近CD19を標的とした2つの自家CAR-T製剤が米国FDAにより"living drug"として承認された。これらの成功により、固形腫瘍を含む他のがん種への応用が期待され、免疫抑制性の腫瘍微小環境を含む固形腫瘍特有の問題点を克服する試みが進められている。今まで臨床試験の中でしか行われてこなかったCAR-T療法へのアクセスが容易になれば、今後は全体の治療計画におけるCAR-T療法の位置付けが重要性になろう。(著者抄録)

  46. The high NK cell count at diagnosis predicts a good response to IST in aplastic anemia

    NARITA Atsushi, MURAMATSU Hideki, XU Yinyan, HAMADA Motoharu, ICHIKAWA Daisuke, NISHIKAWA Eri, KAWASHIMA Nozomu, OKUNO Yusuke, NISHIO Nobuhiro, YAMAZAKI Hirohito, NAKAO Shinji, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本血液学会学術集会抄録(Web)   Vol. 81st   2019

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  47. 肺高血圧に対してトファシチニブが有効であった乳児期発症I型インターフェロン関連疾患の1例

    片岡伸介, 村松秀城, 若松学, 三輪田俊介, 成田幸太郎, 濱田太立, 村上典寛, 谷口理恵子, 市川大輔, 北澤宏展, 鈴木喬悟, 川島希, 西川英里, 成田敦, 奥野友介, 西尾信博, 小島勢二, 深尾敏幸, 大西秀典, 高橋義行

    日本免疫不全・自己炎症学会総会・学術集会プログラム・抄録集   Vol. 2nd   2019

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  48. Whole-genome mutational analysis of juvenile myelomonocytic leukemia

    奥野友介, 村松秀城, 村上典寛, 川島希, 若松学, 北澤宏展, 荻朋男, 高橋義行

    日本癌学会学術総会抄録集(Web)   Vol. 78th   2019

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  49. Analysis of 13 children with hemodiafiltration after hematopoietic stem cell transplantation

    西川英里, 片岡伸介, 濱田太立, 三輪田俊介, 成田幸太郎, 北澤宏展, 村上典寛, 市川大輔, 谷口理恵子, 鈴木喬悟, 川島希, 成田敦, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 41st   2019

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  50. 造血細胞移植後小児における予防接種再開・接種後抗体価に影響する因子の解析

    西川英里, 鈴木高子, 市川大輔, 濱田太立, 川島希, 成田敦, 奥野友介, 村松秀城, 西尾信博, 鳥居ゆか, 川田潤一, 小島勢二, 高橋義行, 伊藤嘉規

    日本ワクチン学会学術集会プログラム・抄録集   Vol. 23rd   2019

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  51. 強度減弱前処置により造血幹細胞移植を施行した原発性免疫不全症の検討

    片岡伸介, 村松秀城, 若松学, 三輪田俊介, 成田幸太郎, 濱田太立, 村上典寛, 谷口理恵子, 市川大輔, 北澤宏展, 鈴木喬悟, 川島希, 西川英里, 奥野友介, 西尾信博, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 41st   2019

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  52. Detection of subclonal SETBP1 and JAK3 mutations in patients with JMML using droplet digital PCR

    WAKAMATSU Manabu, MURAKAMI Norihiro, OKUNO Yusuke, MIWATA Shunsuke, KITAZAWA Hironobu, NARITA Kotaro, KATAOKA Shinsuke, ICHIKAWA Daisuke, HAMADA Motoharu, TANIGUCHI Rieko, KAWASHIMA Nozomu, NISHIKAWA Eri, NARITA Atsushi, NISHIO Nobuhiro, MURAMATSU Hideki, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本血液学会学術集会抄録(Web)   Vol. 81st   2019

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  53. Successful treatment with ATRA + ATO for therapy-related APL caused after Hodgkin’s lymphoma therapy

    YAMAMORI Ayako, NARITA Atsushi, KATAOKA Shinsuke, IMAYA Masayuki, WAKAMATSU Manabu, YOSHIDA Taro, MIWATA Shunsuke, NARITA Kotaro, KITAZAWA Hironobu, TANIGUCHI Rieko, ICHIKAWA Daisuke, HAMADA Motoharu, KAWASHIMA Nozomu, NISHIKAWA Eri, OKUNO Yusuke, NISHIO Nobuhiro, MURAMATSU Hideki, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本血液学会学術集会抄録(Web)   Vol. 81st   2019

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  54. Clinical target sequencing of 28 patients with dyskeratosis congenita

    HAMADA Motoharu, MURAMATSU Hideki, OKUNO Yusuke, MIWATA Shunsuke, NARITA Atsushi, NARITA Kotaro, KITAZAWA Hironobu, ICHIKAWA Daisuke, TANIGUCHI Rieko, KAWASHIMA Nozomu, NISHIKAWA Eri, NISHIO Nobuhiro, NISHIO Nobuhiro, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本血液学会学術集会抄録(Web)   Vol. 81st   2019

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  55. Third party derived EBV specific CTL for treatment of LPD after allogeneic stem cell transplantation

    NISHIO Nobuhiro, NISHIO Nobuhiro, TERAKURA Seitaro, NISHIDA Tetsuya, MURATA Makoto, NISHIKAWA Eri, KAWASHIMA Nozomu, OKUNO Yusuke, OKUNO Yusuke, NARITA Atsushi, MURAMATSU Hideki, KATO Katsuyoshi, KOJIMA Seiji, KIYOI Hitoshi, TAKAHASHI Yoshiyuki

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 41st   2019

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  56. Anti-epiplakin antibodies in bronchiolitis obliterans syndrome after allogeneic stem cell transplant

    川島希, 西川英里, 土坂享成, 室慶直, 橋本隆, 三輪田俊介, 成田幸太郎, 片岡伸介, 濱田太立, 村上典寛, 市川大輔, 鈴木喬悟, 北澤宏展, 谷口理恵, 成田敦, 奥野友介, 西尾信博, 村松秀城, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 41st   2019

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  57. SMARCA4不活化により発症し治療抵抗性を示した胸部腫瘍の1例

    市川大輔, 村松秀城, 奥野友介, 若松学, 三輪田俊介, 成田幸太郎, 北澤宏展, 濱田太立, 片岡伸介, 村上典寛, 谷口理恵子, 鈴木喬悟, 西川英里, 西尾信博, 川島希, 成田敦, 小島勢二, 高橋義行, 中黒匡人, 中村栄男, 中澤温子

    日本小児血液・がん学会雑誌(Web)   Vol. 56 ( 2 )   2019

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  58. Retrospective analysis of 23 children transplanted from HLA 7/8 matched unrelated BMT using ATG

    MURMATSU Hideki, WAKAMATSU Manabu, MIWATA Shunsuke, NARITA Koutarou, HAMADA Motoharu, KATAOKA Shinsuke, MURAKAMI Norihiro, TANIGUCHI Rieko, ICHIKAWA Daisuke, KITAZAWA Hironobu, SUZUKI Kyogo, KAWASHIMA Nozomu, NISHIKAWA Eri, OKUNO Yusuke, NISHIO Nobuhiro, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 41st   2019

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  59. ECMO長期管理から生体肺移植へとつなぎ救命し得た同種骨髄移植後重症肺炎の1例

    村上典寛, 鈴木喬悟, 沼口敦, 池山貴也, 小林求, 濱麻人, 濱田太立, 関屋由子, 堀場千尋, 三輪田俊介, 北澤宏展, 成田幸太郎, 市川大輔, 片岡伸介, 谷口理恵子, 川島希, 西川英里, 奥野友介, 成田敦, 村松秀城, 西尾信博, 小島勢二, 伊藤嘉規, 松田直之, 大藤剛宏, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 41st   2019

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  60. Relation of high expression of p53 in children with Fanconi anemia and the paucity of TP53 mutations

    KAWASHIMA Nozomu, KATAOKA Shinsuke, OKUNO Yusuke, MURAMATSU Hideki, ITO Masafumi, WAKAMATSU Manabu, MIWATA Shunsuke, NARITA Kotaro, HAMADA Motoharu, ICHIKAWA Daisuke, KITAZAWA Hironobu, NISHIKAWA Eri, TANIGUCHI Rieko, NARITA Atsushi, NISHIO Nobuhiro, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本血液学会学術集会抄録(Web)   Vol. 81st   2019

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  61. Analysis of the factors affecting revaccination and the antibody titers in children after HSCT

    NISHIKAWA Eri, SUZUKI Takako, WAKAMATSU Manabu, MIWATA Shunsuke, NARITA Kotaro, YAMAMORI Ayako, KITAZAWA Hironobu, TANIGUCHI Rieko, ICHIKAWA Daisuke, HAMADA Motoharu, KAWASHIMA Nozomu, NARITA Atsushi, OKUNO Yusuke, MURAMATSU Hideki, NISHIO Nobuhiro, KAWADA Jyunichi, KOJIMA Seiji, ITO Yoshinori, TAKAHASHI Yoshiyuki

    日本血液学会学術集会抄録(Web)   Vol. 81st   2019

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  62. Evaluation of complications in cord blood transplantation from KIR ligand incompatible donor

    NARITA Atsushi, MIWATA Shunsuke, NARITA Kotaro, KITAZAWA Hironobu, KATAOKA Shinsuke, HAMADA Motoharu, TANIGUCHI Rieko, ICHIKAWA Daisuke, MURAKAMI Norihiro, SUZUKI Kyogo, NISHIKAWA Eri, KAWASHIMA Nozomu, OKUNO Yusuke, MURAMATSU Hideki, NISHIO Nobuhiro, KOJIMA Seiji, ITO Yoshinori, TAKAHASHI Yoshiyuki

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 41st   2019

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  63. Depletion of monocytes and addition of auto serum improve production of PiggyBac CD19CAR-T cells

    NISHIO Nobuhiro, NISHIO Nobuhiro, SUZUKI Satoshi, TERAKURA Seitaro, IMAHASHI Nobuhiko, HAMADA Motoharu, NISHIKAWA Eri, ICHIKAWA Daisuke, KAWASHIMA Nozomu, OKUNO Yusuke, NARITA Atsushi, MURAMATSU Hideki, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本血液学会学術集会抄録(Web)   Vol. 81st   2019

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  64. 【CAR-T細胞療法の現況】急性リンパ性白血病に対するCAR-T細胞療法

    高橋 義行

    Pharma Medica   Vol. 36 ( 11 ) page: 19 - 23   2018.11

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    Language:Japanese   Publisher:(株)メディカルレビュー社  

  65. 造血幹細胞移植後のhuman herpesvirus 6B(HHV-6B)感染と海馬体積変化の検討

    三宅 未紗, 石原 尚子, 三浦 浩樹, 服部 文彦, 鱸 成隆, 高橋 義行, 小島 勢二, 外山 宏, 夏目 淳, 吉川 哲史

    NEUROINFECTION   Vol. 23 ( 2 ) page: 223 - 223   2018.10

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  66. 放射線治療:QOLを考慮した局所治療 本邦における小児がんに対する陽子線治療の現状と今後

    岩田 宏満, 荻野 浩幸, 伊藤 康彦, 村松 幹司, 亀井 美智, 橋本 眞吾, 中嶌 晃一朗, 高橋 義行, 前田 尚子, 芝本 雄太

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 168 - 168   2018.10

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  67. 小児がんと診断された児のきょうだいへ病状説明を行った事例検討

    滝 良梨子, 佐々木 美和, 新家 一輝, 野田 弘実, 西尾 信博, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 391 - 391   2018.10

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  68. 中枢神経浸潤白血病異種移植マウスモデルにおけるpiggyBac CD19 CAR-T細胞の脳室内投与の安全性と有効性の検討

    田中 邦昭, 加藤 格, 田中 美幸, 盛田 大介, 高橋 義行, 梅田 雄嗣, 平松 英文, 中畑 龍俊, 足立 壯一, 滝田 順子, 中沢 洋三

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 347 - 347   2018.10

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  69. 入院している子どものきょうだい支援 「きょうだいの会」実施報告

    金子 太郎, 佐々木 美和, 新家 一輝, 牧田 夏美, 萩原 沙織, 滝 良梨子, 野田 弘実, 森本 綾, 浅野 みどり, 奈良間 美保, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 390 - 390   2018.10

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  70. 妊娠・子育て中の女性医師が仕事と家庭の両立ができる勤務体制の模索と試み

    高橋 義行

    日本臨床外科学会雑誌   Vol. 79 ( 増刊 ) page: 273 - 273   2018.10

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  71. AYA世代を迎えた小児血液疾患・腫瘍疾患経験者が患者会に求める役割

    太田 晃嗣, 佐々木 美和, 清水 直子, 秋田 麻衣, 谷口 理恵子, 西川 英里, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 55 ( 4 ) page: 382 - 382   2018.10

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  72. 重症再生不良性貧血に対するウサギATG療法後の免疫再構築(Immune reconstitution following rabbit ATG therapy in patients with severe aplastic anemia)

    成田 敦, 村松 秀城, 徐 銀燕, 西川 英里, 川島 希, 奥野 友介, 西尾 信博, 濱 麻人, 山崎 宏人, 中尾 眞二, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1499 - 1499   2018.9

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  73. 肺高血圧に対してトファシチニブが有効であった乳児発症インターフェロン関連疾患の1例(Effectiveness of tofacitinib against pulmonary hypertension in a case of infantile interfereonopathy)

    片岡 伸介, 村松 秀城, 三輪田 俊介, 成田 幸太郎, 濱田 太立, 村上 典寛, 谷口 理恵子, 市川 大輔, 北澤 宏展, 鈴木 喬悟, 川島 希, 西川 英里, 成田 敦, 奥野 友介, 西尾 信博, 小島 勢二, 深尾 敏幸, 大西 秀典, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1684 - 1684   2018.9

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  74. 視神経浸潤を合併しHLA半合致移植によりレスキューできたALLの一例(ALL complicated with optic nerve infiltration was rescued by urgent HLA-haploidentical SCT)

    三輪田 俊介, 成田 敦, 成田 幸太郎, 北澤 宏展, 村上 典寛, 市川 大輔, 濱田 太立, 片岡 伸介, 谷口 理恵子, 鈴木 喬悟, 小島 大英, 川島 希, 西川 英里, 村松 秀城, 西尾 信博, 奥野 友介, 濱 麻人, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1679 - 1679   2018.9

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  75. 本邦におけるShwachman-Diamond症候群の全国的コホートの構築(Establishment of a nationwide cohort for Shwachman-Diamond syndrome in Japan)

    渡邉 健一郎, 金兼 弘和, 濱端 隆行, 上月 景弘, 梅田 雄嗣, 濱 麻人, 奥野 友介, 村松 秀城, 高橋 義行, 長谷川 大輔, 真部 淳, 小原 明, 伊藤 雅文, 小島 勢二, 伊藤 悦朗

    臨床血液   Vol. 59 ( 9 ) page: 1497 - 1497   2018.9

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  76. 新規tagmentation-assisted PCR法を用いたCD19キメラ抗原受容体遺伝子導入T細胞の遺伝子挿入部位解析(Integration mapping of CD19 chimeric antigen receptor T cells by novel tagmentation-assisted PCR)

    濱田 太立, 奥野 友介, 西尾 信博, 鈴木 哲, 川島 希, 村松 秀城, 坪田 庄真, Wilson Matthew H., 盛田 大介, 片岡 伸介, 市川 大輔, 村上 典寛, 谷口 理恵子, 鈴木 喬悟, 小島 大英, 関屋 由子, 西川 英里, 成田 敦, 濱 麻人, 小島 勢二, 中沢 洋三, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1586 - 1586   2018.9

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  77. 慢性活動性Epstein-Barrウイルス感染症における網羅的遺伝子解析(Comprehensive genetic analysis of chronic active Epstein-Barr virus infection)

    奥野 友介, 村田 貴之, 佐藤 好隆, 村松 秀城, 伊藤 嘉規, 奥野 達矢, 村上 典寛, 吉田 健一, 澤田 明久, 井上 雅美, 河 敬世, 瀬戸 加大, 大島 孝一, 白石 友一, 千葉 健一, 田中 洋子, 宮野 悟, 成田 洋平, 吉田 全宏, 渡辺 崇広, 五島 典, 川田 潤一, 西田 徹也, 清井 仁, 加藤 省一, 中村 栄男, 森島 聡子, 藤原 成悦, 清水 則夫, 磯部 泰司, 野口 雅章, 菊田 敦, 岩月 啓氏, 高橋 義行, 小島 勢二, 小川 誠司, 木村 宏

    臨床血液   Vol. 59 ( 9 ) page: 1484 - 1484   2018.9

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  78. 小児非ダウン症急性巨核芽球性白血病における融合遺伝子スクリーニングの臨床的意義(Clinical relevance of fusion gene screening in pediatric AMKL without Down syndrome)

    鈴木 喬悟, 濱 麻人, 奥野 友介, 川島 希, 西川 英里, 成田 敦, 坂口 大俊, 吉田 奈央, 村松 秀城, 西尾 信博, 加藤 剛二, 小島 勢二, Yoo Keon Hee, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1506 - 1506   2018.9

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  79. ダウン症新生児における次世代シーケンサーによるGATA1変異解析(GATA1 mutational analysis using next-generation sequencing in neonates with Down syndrome)

    市川 大輔, 濱 麻人, 奥野 友介, 村松 秀城, 川島 希, 西川 英里, 成田 敦, 西尾 信博, 佐藤 義朗, 立花 貴史, 松沢 要, 早川 昌弘, 吉川 哲史, 奥村 彰久, 齋藤 伸治, 夏目 淳, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1753 - 1753   2018.9

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  80. ファンコニ貧血患者における骨髄の組織病理像とp53発現(Bone marrow histopathology and p53 expression in patients with Fanconi anemia)

    川島 希, 伊藤 雅文, 濱 麻人, 奥野 友介, 村松 秀城, 三輪田 俊介, 成田 幸太郎, 片岡 伸介, 濱田 太立, 村上 典寛, 小島 大英, 市川 大輔, 鈴木 喬悟, 北澤 宏展, 西川 英里, 谷口 理恵子, 成田 敦, 西尾 信博, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1497 - 1497   2018.9

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  81. 低線量放射線とT細胞除去が非腫瘍性疾患に対する骨髄非破壊的前処置を用いた造血細胞移植に与える影響(Impact of low-dose irradiation and TCD in HSCT against non-malignant diseases using a RIC)

    梅田 雄嗣, 矢部 普正, 加藤 剛二, 今井 耕輔, 小林 正夫, 高橋 義行, 井上 雅美, 橋井 佳子, 熱田 由子, 森尾 友宏

    臨床血液   Vol. 59 ( 9 ) page: 1555 - 1555   2018.9

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  82. 小児後天性再生不良性貧血の治療アルゴリズム 日本小児再生不良性貧血治療研究会より(Updated treatment algorithm for children with acquired aplastic anemia in Japan)

    吉田 奈央, 成田 敦, 村松 秀城, 小林 良二, 矢部 普正, 工藤 寿子, 谷ヶ崎 博, 渡邉 健一郎, 森本 哲, 伊藤 悦朗, 大賀 正一, 小原 明, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1626 - 1626   2018.9

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  83. 小児血液腫瘍疾患に合併した非結核性抗酸菌症の4例(Four cases of childhood hematologic disease complicated with nontuberculous mycobacteria infection)

    西川 英里, 村松 秀城, 三輪田 俊介, 成田 幸太郎, 片岡 伸介, 濱田 太立, 北澤 宏展, 村上 典寛, 市川 大輔, 谷口 理恵子, 小島 大英, 鈴木 喬悟, 川島 希, 成田 敦, 奥野 友介, 西尾 信博, 濱 麻人, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1607 - 1607   2018.9

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  84. Epstein-Barrウイルス関連血液悪性疾患における欠損EBVの存在(The presence of defective Epstein-Barr virus(EBV) infection in EBV-associated hematological malignancies)

    奥野 友介, 村田 貴之, 佐藤 好隆, 伊藤 嘉規, 吉田 健一, 澤田 明久, 白石 友一, 宮野 悟, 高橋 義行, 小島 勢二, 小川 誠司, 木村 宏

    日本癌学会総会記事   Vol. 77回   page: 980 - 980   2018.9

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  85. EBV移植後リンパ増殖症に対するテトラマー解析とウイルス特異的CTL療法の有用性(Usefulness of tetramer assay and adoptive transfer of EBV specific CTLs to manage PTLD)

    西尾 信博, 三輪田 俊介, 成田 幸太郎, 北澤 宏展, 片岡 伸介, 濱田 太立, 市川 大輔, 谷口 理恵子, 村上 典寛, 鈴木 喬悟, 川島 希, 西川 英里, 奥野 友介, 成田 敦, 村松 秀城, 鈴木 律朗, 森 毅彦, 川端 浩, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 9 ) page: 1587 - 1587   2018.9

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  86. 若年性骨髄単球性白血病(JMML)に対する包括的遺伝子解析

    村上 典寛, 奥野 友介, 鈴木 喬悟, 成田 敦, 川島 希, 王 希楠, 徐 銀燕, 濱 麻人, 小島 勢二, 村松 秀城, 高橋 義行, 吉田 健一, 真田 昌, 小川 誠司, 白石 友一, 永江 玄太, 千葉 健一, 田中 洋子, 上野 敏秀, 油谷 浩幸, 間野 博行, 宮野 悟, 坂口 大俊, 平山 雅士, 渡辺 新

    日本小児科学会雑誌   Vol. 122 ( 8 ) page: 1398 - 1398   2018.8

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  87. 同種骨髄移植6ヵ月後に再発しCD19-CAR-T療法により1年6ヵ月寛解が得られているMLL-AF4乳児白血病

    西尾 信博, 片岡 伸介, 濱田 太立, 川島 希, 西川 英里, 奥野 友介, 成田 敦, 村松 秀城, 濱 麻人, 小島 勢二, 高橋 義行

    臨床血液   Vol. 59 ( 8 ) page: 1116 - 1116   2018.8

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  88. HLA拘束性のバリアを超えた第3者由来EBV特異的CTL療法の試み

    西尾 信博, 三輪田 俊介, 成田 幸太朗, 北澤 宏展, 濱田 太立, 片岡 伸介, 市川 大輔, 谷口 理恵子, 村上 典寛, 鈴木 喬悟, 西川 英里, 成田 敦, 川島 希, 奥野 友介, 村松 秀城, 濱 麻人, 小島 勢二, 高橋 義行

    MHC: Major Histocompatibility Complex   Vol. 25 ( 2 ) page: 132 - 132   2018.8

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  89. 集学的治療により髄膜播種から完全寛解を達成したAT/RTの一例

    芝 良樹, 近藤 五郎, 栗本 路弘, 夏目 敦至, 若林 俊彦, 大宝 和博, 村松 秀城, 川島 希, 小島 大英, 片岡 伸介, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 55 ( 2 ) page: 211 - 211   2018.7

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  90. 多発皮下腫瘤を呈した11歳男児

    三輪田 俊介, 鈴木 喬悟, 成田 幸太郎, 北澤 宏展, 村上 典寛, 市川 大輔, 濱田 太立, 片岡 伸介, 谷口 理恵子, 小島 大英, 川島 希, 西川 英里, 成田 敦, 村松 秀城, 西尾 信博, 奥野 友介, 濱 麻人, 小島 勢二, 高橋 義行, 岩淵 英人

    日本小児血液・がん学会雑誌   Vol. 55 ( 2 ) page: 209 - 210   2018.7

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  91. 入院している子供のきょうだい支援 「きょうだいの会」実施報告

    金子 太郎, 野田 弘美, 佐々木 美和, 牧田 夏美, 萩原 沙織, 新家 一輝, 浅野 みどり, 奈良間 美保, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 55 ( 1 ) page: 72 - 72   2018.6

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  92. 入院している子供のきょうだい支援 「きょうだいの会」実施報告

    金子 太郎, 野田 弘美, 佐々木 美和, 牧田 夏美, 萩原 沙織, 新家 一輝, 浅野 みどり, 奈良間 美保, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 55 ( 1 ) page: 72 - 72   2018.6

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  93. 「居住地校交流制度」を利用して、入退院を繰り返す治療の中、院内学級と前籍校にて、学校教育を継続できた中学生事例からの一考察

    萩原 沙織, 佐々木 美和, 山田 佳織, 家崎 志のぶ, 近藤 五郎, 村松 秀城, 濱 麻人, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 55 ( 1 ) page: 73 - 74   2018.6

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  94. 胎児期の脳室拡大・大槽拡大が一過性であった先天性サイトメガロウイルス感染症の1例

    田中 雅人, 上田 一仁, 田中 亮, 呉 尚治, 三浦 良介, 浅田 英之, 北瀬 悠磨, 鈴木 俊彦, 立花 貴史, 見松 はるか, 伊藤 美春, 齊藤 明子, 村松 友佳子, 佐藤 義朗, 早川 昌弘, 城所 博之, 川田 潤一, 伊藤 嘉規, 夏目 淳, 高橋 義行

    日本小児科学会雑誌   Vol. 122 ( 5 ) page: 940 - 940   2018.5

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  95. 肉眼的血尿で発症し、化学療法抵抗性の経過を示した14歳腎腫瘍の1例

    市川 大輔, 村松 秀城, 成田 幸太郎, 北澤 宏展, 濱田 太立, 片岡 伸介, 村上 典寛, 谷口 理恵子, 小島 大英, 鈴木 喬悟, 西川 英里, 西尾 信博, 奥野 友介, 川島 希, 成田 敦, 濱 麻人, 小島 勢二, 高橋 義行, 内田 広夫, 下山 芳江, 中村 栄男, 山下 享子, 中澤 温子

    日本小児血液・がん学会雑誌   Vol. 54 ( 5 ) page: 440 - 440   2018.4

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  96. 難治性マイコプラズマ肺炎に対するステロイド療法の検討

    田中 雅大, 川田 潤一, 奥村 俊彦, 石黒 智紀, 平山 祐司, 楢原 翔, 辻 元基, 成田 幸太郎, 杉山 裕一朗, 鈴木 道雄, 辻 健史, 星野 伸, 佐藤 義朗, 高橋 義行

    日本小児科学会雑誌   Vol. 122 ( 2 ) page: 335 - 335   2018.2

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  97. 新生児分野で臨床応用が期待される基礎研究 新生児慢性肺疾患(CLD)に対する幹細胞療法

    鈴木 俊彦, 佐藤 義朗, 村松 友佳子, 高橋 義行, 早川 昌弘

    日本小児科学会雑誌   Vol. 122 ( 2 ) page: 194 - 194   2018.2

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  98. 小児ヒトメタニューモウイルス感染症に関する多施設前向き臨床調査

    谷口 顕信, 川田 潤一, 郷 清貴, 藤城 尚純, 細川 洋輔, 牧 祐輝, 杉山 裕一朗, 鈴木 道雄, 辻 健史, 星野 伸, 村松 秀城, 城所 博之, 佐藤 義朗, 夏目 淳, 高橋 義行

    日本小児科学会雑誌   Vol. 122 ( 2 ) page: 300 - 300   2018.2

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  99. 予防可能死を減らすためのチャイルドデスレビュー:医療者は何が出来るか 多機関連携でのCDRの実施の実際 医療者の果たすべき役割

    沼口 敦, 夏目 淳, 早川 昌弘, 伊藤 嘉規, 齋藤 伸治, 吉川 哲史, 奥村 彰久, 岩佐 充二, 池山 貴也, 山崎 嘉久, 高橋 義行

    日本小児科学会雑誌   Vol. 122 ( 2 ) page: 204 - 204   2018.2

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  100. shuffling babyの神経発達予後

    岡井 佑, 三浦 清邦, 若子 理恵, 高橋 脩, 田中 雅大, 坂口 陽子, 伊藤 祐史, 山本 啓之, 大野 敦子, 中田 智彦, 城所 博之, 高橋 義行, 夏目 淳

    日本小児科学会雑誌   Vol. 122 ( 2 ) page: 370 - 370   2018.2

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  101. 胎児鏡下気管閉塞術を施行後に重度の気管軟化を呈した先天性横隔膜ヘルニアの1例

    加藤 俊輔, 高橋 義行, 田中 雅人, 上田 一仁, 田中 亮, 三浦 良介, 呉 尚治, 浅田 英之, 北瀬 悠磨, 鈴木 俊彦, 立花 貴史, 見松 はるか, 伊藤 美春, 齊藤 明子, 村松 友佳子, 佐藤 義朗, 早川 昌弘

    日本小児科学会雑誌   Vol. 122 ( 1 ) page: 113 - 113   2018.1

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  102. 当院の小児がんに対する陽子線治療の実績と管理体制

    武田 理沙, 鈴木 博乃, 水野 奈々, 多湖 明香里, 吉田 明生, 伊藤 翠, 福田 恵子, 側島 健宏, 山田 拓司, 横井 暁子, 小林 悟, 神岡 直美, 濱嶋 直樹, 村松 幹司, 鈴木 悟, 後藤 玄夫, 戸苅 創, 亀井 美智, 伊藤 康彦, 高橋 義行, 前田 尚子

    日本小児科学会雑誌   Vol. 122 ( 1 ) page: 117 - 118   2018.1

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  103. 集学的治療により髄膜播種から完全寛解を達成したAT/RTの一例

    芝良樹, 近藤五郎, 栗本路弘, 夏目敦至, 若林俊彦, 大宝和博, 村松秀城, 川島希, 小島大英, 片岡伸介, 高橋義行

    日本小児血液・がん学会雑誌(Web)   Vol. 55 ( 2 )   2018

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  104. 多発皮下腫瘤を呈した11歳男児

    三輪田俊介, 鈴木喬悟, 成田幸太郎, 北澤宏展, 村上典寛, 市川大輔, 濱田太立, 片岡伸介, 谷口理恵子, 小島大英, 川島希, 西川英里, 成田敦, 村松秀城, 西尾信博, 奥野友介, 濱麻人, 小島勢二, 高橋義行, 岩淵英人

    日本小児血液・がん学会雑誌(Web)   Vol. 55 ( 2 )   2018

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  105. 入院中の中高生を対象とした、大学生による学習支援ボランティアの取り組みの現状と課題

    篠原 夏美, 佐々木 美和, 森本 綾, 野田 弘実, 村松 秀城, 高橋 義行, 浅野 みどり, 奈良間 美保

    日本小児血液・がん学会雑誌   Vol. 54 ( 3 ) page: 271 - 271   2017.12

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  106. Risk Factors for Donor-Type Aplasia after Bone Marrow Transplant in Children with Acquired Bone Marrow Failure

    Asahito Hama, Hideki Muramatsu, Atsushi Narita, Hironobu Kitazawa, Motoharu Hamada, Shinsuke Kataoka, Norihiro Murakami, Daisuke Ichikawa, Rieko Taniguchi, Daiei Kojima, Kyogo Suzuki, Eri Nishikawa, Yusuke Okuno, Nozomu Kawashima, Nobuhiro Nishio, Masafumi Ito, Seiji Kojima, Yoshiyuki Takahashi

    BLOOD   Vol. 130   2017.12

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    Web of Science

  107. Cited2はCBP/p300-beta-catenin-Sox9を介して小児悪性固形腫瘍である神経芽腫の腫瘍形成と転移を抑制する

    岸田 聡, 曹 東亮, 成田 敦, 吉田 安子, 高橋 義行, 門松 健治

    生命科学系学会合同年次大会   Vol. 2017年度   page: [2P - 0892]   2017.12

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  108. Clinical Sequencing of 347 Children with Acquired and Inherited Bone Marrow Failure Syndromes

    Hideki Muramatsu, Yusuke Okuno, Kenichi Yoshida, Yuichi Shiraishi, Sayoko Doisaki, Atsushi Narita, Hirotoshi Sakaguchi, Nozomu Kawashima, Xinan Wang, Yinyan Xu, Kenichi Chiba, Hiroko Tanaka, Asahito Hama, Masashi Sanada, Yoshiyuki Takahashi, Hitoshi Kanno, Hiroki Yamaguchi, Shouichi Ohga, Atsushi Manabe, Hideo Harigae, Shinji Kunishima, Eiichi Ishii, Masao Kobayashi, Kenichi Koike, Kenichiro Watanabe, Etsuro Ito, Minoru Takata, Miharu Yabe, Seishi Ogawa, Satoru Miyano, Seiji Kojima

    BLOOD   Vol. 130   2017.12

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  109. Durable Remission after Salvage Chemotherapy with Combining Bortezomib and Vorinostat Followed By CD19 CAR-T Therapy in a Patient with MLL-Rearranged ALL after Early Relapse Post Allogeneic Hematopoietic Stem Cell Transplantation

    Nobuhiro Nishio, Kai Wang, Kotaro Narita, Shinsuke Kataoka, Hironobu Kitazawa, Motoharu Hamada, Daisuke Ichikawa, Rieko Taniguchi, Norihiro Murakami, Daiei Kojima, Kyogo Suzuki, Nozomu Kawashima, Eri Nishikawa, Yusuke Okuno, Atsushi Narita, Hideki Muramatsu, Asahito Hama, Shih-Ting Tsao, Maoquan Qin, Huyong Zheng, Lung-Ji Chang, Seiji Kojima, Yoshiyuki Takahashi

    BLOOD   Vol. 130   2017.12

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  110. 当院における小児長期フォローアップ外来の取り組み(第一報)

    清水 直子, 秋田 麻衣, 佐々木 美和, 滝 良梨子, 野田 弘実, 西川 英里, 谷口 理恵子, 高橋 義行

    日本小児血液・がん学会雑誌   Vol. 54 ( 4 ) page: 447 - 447   2017.10

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  111. ATGを用いた造血幹細胞移植 予防と治療

    西尾 信博, 高橋 義行, 小島 勢二

    臨床血液   Vol. 58 ( 10 ) page: 2104 - 2110   2017.10

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    同種造血幹細胞移植後のgraft versus host disease(GVHD)は移植関連合併症,移植関連死亡を増加させ,quality of life(QOL)を低下させる。一方,GVHDの発症を抑えるための過度な免疫抑制は移植後感染症や悪性疾患の再発を増加させるため,移植の成功のためには適切なGVHD予防が求められる。Anti-T cell globulin(ATG)はカルシニューリン阻害薬やメソトレキセートなどの免疫抑制剤との併用により,重度の感染症や悪性疾患の再発を増加させることなく,重症の急性GVHDや全身型慢性GVHDの発症を低下させる。ATGの血中動態は患者間で大きく異なり,このことは移植結果に影響を与えるため,患者ごとに必要な投与量を予測するための研究が必要である。ステロイド抵抗性GVHDの治療薬としてのATGの位置付けは限定的であり,今後の研究が待たれる。(著者抄録)

  112. Congenetal dyserythropoietic anemia 10例の全エクソーム解析結果(Whole exome sequency analysis of congenital dyserythropoietic anemia in Japan)

    Hamada Motoharu, Doisaki Sayoko, Okuno Yusuke, Muramatsu Hideki, Hama Asahito, Kataoka Shinsuke, Ichikawa Daisuke, Taniguchi Rieko, Suzuki Kyogo, Kojima Daiei, Sekiya Yuko, Kawashima Nozomu, Narita Atsushi, Yoshida Kenichi, Shiraishi Yuichi, Sanada Masashi, Chiba Kenichi, Tanaka Hiroko, Manabe Atsushi, Taga Takashi, Takahashi Yoshiyuki, Miyano Satoru, Ogawa Seishi, Kojima Seiji

    臨床血液   Vol. 58 ( 9 ) page: 1477 - 1477   2017.9

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    J-GLOBAL

  113. 介護予防事業を体育協会が委託を受けての課題と対策

    森岡 淳, 吉川 理沙, 前川 政人, 高橋 迪子, 石山 美貴, 菅 義行

    日本転倒予防学会誌   Vol. 4 ( 2 ) page: 72 - 72   2017.9

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  114. 非血縁者間臍帯血移植後に焦点性発作、海馬の異常が見られHHV-6の再活性化が疑われた1例

    田中 雅大, 岡井 佑, 坂口 陽子, 伊藤 祐史, 山本 啓之, 大野 敦子, 中田 智彦, 城所 博之, 川田 潤一, 伊藤 嘉規, 濱 麻人, 村松 秀城, 根来 民子, 渡邊 一功, 高橋 義行, 夏目 淳

    日本小児科学会雑誌   Vol. 121 ( 8 ) page: 1439 - 1440   2017.8

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  115. 愛知県における重症複合免疫不全症とポンペ病に対する新生児マススクリーニング導入

    小島 大英, 奥野 友介, 村松 秀城, 小島 勢二, 高橋 義行, 中島 葉子, 伊藤 哲哉, 柘植 郁哉, 酒井 好美, 加藤 智章

    日本小児科学会雑誌   Vol. 121 ( 7 ) page: 1270 - 1270   2017.7

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  116. 大学病院と市中病院小児科が連携し在宅で終末期を過ごした小児脳腫瘍症例

    大辻 塩見, 山内 悠太郎, 小早川 雄介, 浅井 慎平, 服部 亜季子, 山口 直哉, 田村 泉, 安井 竜志, 中嶋 枝里子, 宮地 利英子, 後藤 紀子, 浅井 雅美, 水野 美穂子, 近藤 五郎, 栗本 路弘, 村松 秀城, 片岡 伸介, 高橋 義行

    日本小児科学会雑誌   Vol. 121 ( 7 ) page: 1275 - 1275   2017.7

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  117. 本邦における肝芽腫に対する造血幹細胞移植を用いた大量化学療法の治療成績

    山本 将平, 渡邊 健一郎, 井上 雅美, 橋井 佳子, 菊田 敦, 金子 隆, 加藤 剛二, 原 純一, 田渕 健, 高橋 義行, 日本造血細胞移植学会移植一元化固形腫瘍委員会

    日本小児血液・がん学会雑誌   Vol. 54 ( 2 ) page: 126 - 132   2017.6

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    肝芽腫の治療成績は化学療法、手術、および肝移植により改善したが、遠隔転移や肝門部病変などの切除不能病変を有する症例の治療成績は依然として不良である。これらの症例に対して造血幹細胞移植を用いた大量化学療法が施行されることもあるがその有効性は明らかでない。そこで、今回、日本造血細胞移植学会造血細胞移植登録一元管理委員会に造血幹細胞移植を受け登録された、肝芽腫136例(のべ移植回数162回)の治療成績について後方視的に検討した。移植病期は寛解期70例、非寛解期80例、不明12例であった。前処置HiMEC(CBDCA、VP16、L-PAM)、TEPA+L-PAMが最も多くそれぞれ51、43例であった。全体の5年全生存率、5年無病生存率はそれぞれ63.7%、50.3%であった。移植時寛解群と非寛解群の比較では5年全生存率でそれぞれ80.2%、49.8%(p<0.05)、5年無病生存率でそれぞれ62.4%、38.4%(p<0.05)であり非寛解群において有意に低かった。しかし、移植時非寛解群においても5年全生存率は50%弱であり過去の報告と同程度であった。肝移植、外科的治療の進歩により肝芽腫に対する大量化学療法は行われなくなってきているが、今後、大量化学療法の有効性を評価する場合には、診断時遠隔転移症例など再発リスクの高い症例に対し、統一レジメンを用いた臨床試験を行ってなされる必要があると思われる。(著者抄録)

  118. 抗GD2抗体治療により大量化学療法後の残存腫瘍の縮小と腫瘍マーカーの低下が得られた難治性ハイリスク神経芽腫の1例

    濱田 太立, 成田 敦, 北澤 宏展, 片岡 伸介, 市川 大輔, 村上 典寛, 谷口 理恵子, 小島 大英, 鈴木 喬悟, 関屋 由子, 川島 希, 西川 英里, 西尾 信博, 村松 秀城, 濱 麻人, 小島 勢二, 高橋 義行, 申 将守, 今井 千速

    日本小児血液・がん学会雑誌   Vol. 54 ( 2 ) page: 175 - 175   2017.6

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  119. 嚢胞性部分的分化型腎芽腫の一例

    片岡 伸介, 村松 秀城, 北澤 宏展, 濱田 太立, 市川 大輔, 村上 典寛, 小島 大英, 鈴木 喬悟, 関屋 由子, 西川 英里, 西尾 信博, 奥野 友介, 川島 希, 成田 敦, 濱 麻人, 小島 勢二, 高橋 義行, 田中 裕次郎, 内田 広夫, 下山 芳江, 中黒 匡人, 中村 栄男

    日本小児血液・がん学会雑誌   Vol. 54 ( 2 ) page: 173 - 173   2017.6

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  120. PiggyBac遺伝子改変CD19キメラ抗原受容体T細胞療法のFirst in Human試験にむけた基盤研究

    西尾 信博, 鈴木 哲, 奥野 友介, 中沢 洋三, 田中 美幸, 盛田 大介, 濱田 太立, 川島 希, 成田 敦, 村松 秀城, 濱 麻人, 小島 勢二, 高橋 義行

    臨床血液   Vol. 58 ( 6 ) page: 687 - 687   2017.6

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  121. 造血幹細胞移植を受けた小児における白質障害 拡散テンソル画像解析

    坂口 陽子, 田中 雅大, 岡井 佑, 伊藤 祐史, 山本 啓之, 大野 敦子, 中田 智彦, 城所 博之, 高橋 義行, 夏目 淳

    脳と発達   Vol. 49 ( Suppl. ) page: S341 - S341   2017.5

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  122. 小児固形腫瘍における病理組織検査と融合遺伝子検査での診断不一致例の検討

    市川 大輔, 村松 秀城, 奥野 友介, 濱田 太立, 片岡 伸介, 谷口 理恵子, 村上 典寛, 鈴木 喬悟, 小島 大英, 関屋 由子, 西川 英里, 川島 希, 成田 敦, 西尾 信博, 濱 麻人, 小島 勢二, 下山 芳江, 中村 栄男, 高橋 義行

    日本小児科学会雑誌   Vol. 121 ( 4 ) page: 789 - 789   2017.4

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  123. 診断に苦慮した胸腔腫瘍の男児例

    村上 典寛, 濱 麻人, 市川 大輔, 西川 英里, 濱田 太立, 片岡 伸介, 谷口 理恵子, 小島 大英, 鈴木 喬悟, 関屋 由子, 川島 希, 成田 敦, 村松 秀城, 西尾 信博, 奥野 友介, 小島 勢二, 高橋 義行, 島田 聡子, 下山 芳江, 中村 栄男

    日本小児血液・がん学会雑誌   Vol. 53 ( 5 ) page: 491 - 491   2017.3

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  124. 発熱性好中球減少症に対するカルバペネム・ミカファンギン併用初期治療の有用性

    西川 英里, 関屋 由子, 川島 希, 奥野 友介, 成田 敦, 村松 秀城, 西尾 信博, 濱 麻人, 小島 勢二, 高橋 義行

    日本小児科学会雑誌   Vol. 121 ( 2 ) page: 291 - 291   2017.2

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  125. 捻転した虫垂粘液腫の1例

    佐々木 陽子, 下條 紋季, 折戸 信暁, 山田 篤史, 高橋 孝博, 左合 直, 川上 義行, 太田 諒

    Japanese Journal of Radiology   Vol. 35 ( Suppl. ) page: 33 - 33   2017.2

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  126. 愛知県における小児死因調査の試み

    沼口 敦, 高橋 義行, 夏目 淳, 伊藤 嘉規, 大野 敦子, 齋藤 伸治, 奥村 彰久, 吉川 哲史

    日本小児科学会雑誌   Vol. 121 ( 2 ) page: 334 - 334   2017.2

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  127. 小児菌血症患者の病原菌診断における次世代シーケンサーの臨床応用

    堀場 千尋, 川田 潤一, 手塚 宜行, 鈴木 高子, 安藤 将太郎, 神谷 泰子, 鳥居 ゆか, 高橋 義行, 伊藤 嘉規

    日本小児科学会雑誌   Vol. 121 ( 2 ) page: 242 - 242   2017.2

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  128. ここまで進歩した小児白血病・がんに対する治療法 小児造血細胞移植の進歩と展望

    高橋 義行

    日本小児科学会雑誌   Vol. 121 ( 2 ) page: 219 - 219   2017.2

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  129. 【骨髄不全症の最近の進歩】先天性骨髄不全症のゲノム診断

    村松 秀城, 小島 勢二, 高橋 義行

    血液フロンティア   Vol. 27 ( 2 ) page: 213 - 218   2017.1

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    先天性骨髄不全症は,患者の有する遺伝子変異が原因となり造血不全を合併する症候群の総称であり,多数の疾患を含んでいる。最近のゲノム解析技術の向上により,先天性骨髄不全症の新規原因遺伝子が次々と報告されている。一方,先天性骨髄不全症の確定診断には,遺伝子変異解析が有用だが,疾患数および原因遺伝子が多数存在するため実際には多くの困難を伴う。近年,次世代シーケンサーを用いた網羅的遺伝子診断システムが構築され,多数の疾患の原因遺伝子を同時に解析・評価することが技術的に可能となっており,臨床での広範な利用が期待される。(著者抄録)

  130. 【貧血学-最新の診断・治療動向-】骨髄疾患による貧血 先天性角化不全症 先天性角化不全症の疾患概念・病因・病態

    西尾 信博, 高橋 義行

    日本臨床   Vol. 75 ( 増刊1 貧血学 ) page: 422 - 426   2017.1

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  131. 【貧血学-最新の診断・治療動向-】骨髄疾患による貧血 先天性角化不全症 先天性角化不全症の検査・診断・治療

    西尾 信博, 高橋 義行

    日本臨床   Vol. 75 ( 増刊1 貧血学 ) page: 427 - 430   2017.1

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  132. Risk factor for donor-type aplasia after bone marrow transplant in children with bone marrow failure

    HAMA Asahito, MURAMATSU Hideki, NARITA Atsushi, KITAZAWA Hironobu, HAMADA Motoharu, KATAOKA Shinsuke, MURAKAMI Norihiro, ICHIKAWA Daisuke, TANIGUCHI Rieko, KOJIMA Daiei, SUZUKI Kyogo, NISHIKAWA Eri, OKUNO Yusuke, KAWASHIMA Nozomu, NISHIO Nobuhiro, ITO Masafumi, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    臨床血液   Vol. 58 ( 9 )   2017

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  133. Efficacy of carbapenem combined with micafungin for blood stream infections with febrile neutropenia

    NISHIKAWA Eri, SEKIYA Yuko, KATAOKA Shinsuke, HAMADA Motoharu, ICHIKAWA Daisuke, KOJIMA Daiei, MURAKAMI Norihiro, KITAZAWA Hironobu, TANIGUCHI Rieko, SUZUKI Kyogo, KAWASHIMA Nozomu, OKUNO Yusuke, NARITA Atsushi, MURAMATSU Hideki, NISHIO Nobuhiro, HAMA Asahito, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    臨床血液   Vol. 58 ( 9 )   2017

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  134. 移植後蛋白漏出性胃腸症による低IgG血症に皮下注用人免疫グロブリン製剤の補充療法が有用であった1例

    三輪田俊介, 小島大英, 村松秀城, 成田幸太郎, 北澤宏展, 片岡伸介, 濱田太立, 谷口理恵子, 村上典寛, 市川大輔, 鈴木喬悟, 川島希, 西川英里, 成田敦, 奥野友介, 西尾信博, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 40th   2017

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  135. 肉眼的血尿で発症し,化学療法抵抗性の経過を示した14歳腎腫瘍の1例

    市川大輔, 村松秀城, 成田幸太郎, 北澤宏展, 濱田太立, 片岡伸介, 村上典寛, 谷口理恵子, 小島大英, 鈴木喬悟, 西川英里, 西尾信博, 奥野友介, 川島希, 成田敦, 濱麻人, 小島勢二, 高橋義行, 内田広夫, 下山芳江, 中村栄男, 山下享子, 中澤温子

    日本小児血液・がん学会雑誌(Web)   Vol. 54 ( 5 )   2017

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  136. 臍帯血移植を施行したDOCK8欠損症の1例

    片岡伸介, 村松秀城, 小島大英, 濱田太立, 市川大輔, 谷口理恵子, 村上典寛, 鈴木喬悟, 関屋由子, 西川英里, 西尾信博, 奥野友介, 川島希, 成田敦, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  137. MYB rearrangements in biastic plasmacytoid dendritic cell neoplasm

    SUZUKI Kyogo, OKUNO Yusuke, SUZUKI Yuka, HAMA Asahito, MURAMATSU Hideki, NAKATOCHI Masahiro, GUNJI Masaharu, ICHIKAWA Daisuke, HAMADA Motoharu, TANIGUCHI Rieko, KATAOKA Shinsuke, MURAKAMI Norihiro, KOJIMA Daiei, NISHIKAWA Eri, KAWASHIMA Nozomu, NARITA Atsushi, NISHIO Nobuhiro, NAKAZAWA Yozo, IWAFUCHI Hideto, WATANABE Kenichiro, ITO Masafumi, KOJIMA Seiji, KATO Seiichi, TAKAHASHI Yoshiyuki

    臨床血液   Vol. 58 ( 9 )   2017

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  138. 小児造血細胞移植後,肺高血圧症の検討

    川島希, 西川英里, 深澤佳絵, 成田幸太郎, 片岡伸介, 濱田太立, 北澤宏展, 市川大輔, 村上典寛, 小島大英, 鈴木喬悟, 谷口理恵子, 成田敦, 奥野友介, 村松秀城, 西尾信博, 濱麻人, 小島勢二, 加藤太一, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 40th   2017

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  139. FANCD2 monoubiquitination for diagnosing Fanconi anemia in the era of high-throughput sequencing

    KAWASHIMA Nozomu, OKUNO Yusuke, MURAMATSU Hideki, HAMA Asahito, NARITA Atsushi, KATAOKA Shinsuke, HAMADA Motoharu, ICHIKAWA Daisuke, MURAKAMI Norihito, KOJIMA Daiei, SUZUKI Kyogo, NISHIKAWA Eri, NISHIO Nobuhiro, NAKAZAWA Yozo, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    臨床血液   Vol. 58 ( 9 )   2017

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  140. ハイリスク神経芽腫に対するKIRリガンド不一致臍帯血移植後におけるアロ反応性NK細胞数の増加

    西尾信博, 西尾信博, 片岡伸介, 市川大輔, 谷口理恵子, 村上典寛, 小島大英, 鈴木喬吾, 関屋由子, 川島希, 西川英里, 奥野友介, 奥野友介, 成田敦, 村松秀城, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  141. 再発Wilms腫瘍に対する自家末梢血幹細胞移植併用大量化学療法

    関屋由子, 濱麻人, 片岡伸介, 濱田太立, 村上典寛, 市川大輔, 谷口理恵子, 小島大英, 鈴木喬悟, 川島希, 西川英里, 成田敦, 奥野友介, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  142. 包括的遺伝子解析を行った若年性骨髄単球性白血病に対する同種造血細胞移植の成績

    村松秀城, 村上典寛, 奥野友介, 坂口大俊, 吉田健一, 上野敏秀, 白石友一, 千葉健一, 田中洋子, 小島大英, 鈴木喬悟, 川島希, 成田敦, 西尾信博, 濱麻人, 富野悟, 小川誠司, 間野博行, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  143. 同種造血幹細胞移植を実施したIL2Rα欠損症の1例

    小島大英, 村松秀城, 森雄司, 柘植郁哉, 成田幸太郎, 北澤宏展, 片岡伸介, 濱田太立, 谷口理恵子, 村上典寛, 市川大輔, 鈴木喬悟, 川島希, 西川英里, 成田敦, 奥野友介, 西尾信博, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 40th   2017

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  144. Effect of glutathione S-transferase gene variations on busulfan pharmacokinetics in children

    NARITA Atsushi, MIYAZAKI Masayuki, HAMADA Motoharu, KATAOKA Shinsuke, ICHIKAWA Daisuke, TANIGUCHI Rieko, MURAKAMI Norihiro, KOJIMA Daiei, SUZUKI Kyogo, NISHIKAWA Eri, KAWASHIMA Nozomu, OKUNO Yusuke, MURAMATSU Hideki, NISHIO Nobuhiro, HAMA Asahito, SAKAGUCHI Hirotoshi, YOSHIDA Nao, KATO Koji, NAGAI Taku, YAMADA Kiyofumi, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    臨床血液   Vol. 58 ( 9 )   2017

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  145. 小児同種移植後の拒絶後に対する緊急HLAハプロ一致血縁者移植によるレスキュー

    西川英里, 片岡伸介, 濱田太立, 市川大輔, 小島大英, 村上典寛, 鈴木喬吾, 関屋由子, 谷口理恵子, 川島希, 奥野友介, 成田敦, 村松秀城, 西尾信博, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  146. 小児同種造血幹細胞移植後の肝中心静脈閉塞症(VOD)に対するリスク因子解析および遺伝子組み換えトロンボモジュリン(rTM)の安全性と有効性についての検討

    濱田太立, 成田敦, 片岡伸介, 市川大輔, 村上典寛, 谷口理恵子, 小島大英, 鈴木喬吾, 関屋由子, 川島希, 西川英里, 奥野友介, 西尾信博, 村松秀城, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  147. High-dose chemotherapy with autologous stem cell transplantation for pediatric solid tumors

    NISHIKAWA Eri, KATAOKA Shinsuke, HAMADA Motoharu, NARITA Koutarou, MURAKAMI Norihiro, ICHIKAWA Daisuke, KITAZAWA Hironobu, KOJIMA Daiei, SUZUKI Kyogo, TANIGUCHI Rieko, KAWASHIMA Nozomu, OKUNO Yusuke, NARITA Atsushi, MURAMATSU Hideki, NISHIO Nobuhiro, HAMA Asahito, KOJIMA Seiji, TAKAHASHI Yoshiyuki

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 40th   2017

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  148. 小児造血幹細胞移植におけるウイルス感染症のリスク因子

    成田敦, 伊藤嘉規, 西川英里, 川島希, 村松秀城, 西尾信博, 鳥居ゆか, 川田潤一, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  149. 小児造血細胞移植前の腎機能評価:24時間クレアチニン・クリアランス,推算糸球体濾過量,99mTc-DTPA腎動態シンチの比較

    川島希, 片岡伸介, 濱田太立, 市川大輔, 小島大英, 鈴木喬悟, 村上典寛, 谷口理恵子, 関屋由子, 西川英里, 成田敦, 村松秀城, 西尾信博, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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  150. Excellent outcomes of children with CML treated with TKI and allo-HSCT with RIC regimen

    KITAZAWA Hironobu, MURAMATSU Hideki, KATAOKA Shinsuke, HAMADA Motoharu, MURAKAMI Norihiro, ICHIKAWA Daisuke, TANIGUCHI Rieko, KOJIMA Daiei, SUZUKI Kyogo, KAWASHIMA Nozomu, NISHIKAWA Eri, OKUNO Yusuke, NARITA Atsushi, NISHIO Nobuhiro, HAMA Asahito, KOJOMA Seiji, TAKAHASHI Yoshiyuki

    臨床血液   Vol. 58 ( 9 )   2017

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  151. Diamond-Blackfan貧血に対するフルダラビンおよびメルファランを用いた同種骨髄移植

    濱麻人, 成田敦, 鈴木喬悟, 成田幸太郎, 北澤宏展, 濱田太立, 片岡伸介, 村上典寛, 市川大輔, 谷口理恵子, 小島大英, 西川英里, 奥野友介, 川島希, 村松秀城, 西尾信博, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 40th   2017

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  152. Targeted-BU+MELの前処置を用いて同種造血細胞移植を施行したWiskott-Aldrich症候群の3例

    小島大英, 村松秀城, 片岡伸介, 濱田太立, 村上典寛, 市川大輔, 谷口理恵子, 鈴木喬悟, 関屋由子, 川島希, 西川英里, 奥野友介, 成田敦, 西尾信博, 濱麻人, 小島勢二, 高橋義行

    日本造血細胞移植学会総会プログラム・抄録集   Vol. 39th   2017

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KAKENHI (Grants-in-Aid for Scientific Research) 13

  1. 新規神経芽腫マーカー(CTN/VLA)の有効性(診断率・悪性度予測)検証と病態解明

    Grant number:24K11018  2024.4 - 2027.3

    日本学術振興会  科学研究費助成事業  基盤研究(C)

    檜 顕成, 内田 広夫, 高橋 義行, 成田 敦, 大澤 毅, 城田 千代栄, 田井中 貴久, 天野 日出, 牧田 智, 横田 一樹

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    Authorship:Coinvestigator(s) 

    CTN/VLAは既存マーカーと比較し悪性度を予測できる点で臨床的に有効なマーカーになり得る可能性があり、世界的にもCTNとMYC変異との強い相関が注目されつつある。特に最近の研究においてCTNについては、腫瘍細胞フェロトーシスと脂質過酸化(CTN蓄積)、MYC変異との関与から、神経芽腫の自然退縮(細胞死)との関連が示唆され、フェロトーシスの調節が神経芽腫の将来の治療に繋がると考えられている。
    本研究ではMYC遺伝子増幅神経芽腫サンプル/非増幅サンプル/健常コントロールサンプルにおいてCTN/VLAの有効性(診断率・腫瘍悪性度予測)の検証を行う。

  2. Improvement and Validation of a Discriminant Model for Amplification of the Neuroblastoma MYC Gene by Tyrosine Metabolite Markers

    Grant number:23K07308  2023.4 - 2026.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Scientific Research (C)

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  3. 腎芽腫メタボローム解析(尿/組織)による診断マーカーとオンコメタボライト探索

    Grant number:22K07276  2022.4 - 2025.3

    日本学術振興会  科学研究費助成事業  基盤研究(C)

    岡本 眞宗, 城田 千代栄, 成田 敦, 牧田 智, 田井中 貴久, 内田 広夫, 高橋 義行, 大澤 毅, 住田 亙, 田中 裕次郎, 檜 顕成

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    増殖・浸潤・転移などがんの進展における中心代謝と代謝リプログラミングの役割については未だ不明な点が多い。本研究で腎芽腫細胞に特異的な中心代謝に関与する代謝物(オンコメタボライト)を見出す。がん細胞に特異的な代謝系を見出すことにより、正常細胞の代謝系との違いを標的とする新たながんの治療戦略の開発を目指す。腎芽腫患児の尿、腫瘍組織のキャピラリー電気泳動質量分析法を中心とした代謝物解析により、中心代謝に関与するオンコメタボライトを探索し、従来の集学的治療と遺伝子解析だけでは解明できなかった病態解明及び新たな診断治療法の開発に繋げる。
    Wilms腫瘍は小児の腎腫瘍の中で最も頻度が高い腫瘍であり、小児腎腫瘍の90%を占める。現在ではStageⅠであれば90%以上の5年生存率が得られ治癒可能な病気へと変化してきたが、現行の治療法では未だ十分な治療効果とは言えない。腎動脈や大動脈周囲のリンパ節や肺に転移をきたしやすく、腎静脈から下大静脈内に 腫瘍塞栓を形成することもあり、全体の約10%に予後不良群を認める。最近ではメタボロミクス技術の発展により生体内の代謝情報を鋭敏且つ包括的に捉えることが可能となり、様々な癌種において新たなバイオマーカーや病態の解明が進んでいる。
    本研究はWilms腫瘍における患児尿と健常コントロール尿の 代謝プロファイルの違いや関連を明らかにすることを目的とし 、CE/MS:キャピラリー電気泳動質量分析法を中心に代謝物を測定した。(腎芽腫、健常コントロール 計10サンプル)
    質量分析法の種類(CE/MS、LC/MS)により、検出可能な代謝物質の網羅性・定量性が大きく異なることがわかっている。実際にCE/MSはイオン性化合物に対し高い分離能を有するが、実際の測定は煩雑である。一方LC/MSは核酸・脂肪酸・リン酸化合物をはじめとした幅広い測定が可能であるがイオン性物質の分離能は低い。単独の測定法ですべての代謝物質を網羅するのは困難なため、比較的特性の似ている化合物群に対し、適切な分析装置を選択し疾患特異的な代謝物を特定する必要がある。そこでCE/MSに加え、同時にLC/MSでも組織中の代謝物を測定中である。
    順調に腎芽腫尿サンプルと腎芽腫組織サンプルを臨床情報付きで収集し、キャピラリー電気泳動質量分析法(CE/MS)、液体クロマトグラフィー質量分析法(LC/MS)で測定した。現在データ解析中。
    引き続きWilms腫瘍サンプル及びコントロールサンプル(尿・組織)を収集し、Wilms腫瘍において疾患貢献度の高い代謝物をターゲットに、疾病機序の解明、尿中バイオマーカー探索を続けていく。

  4. Investigating the pathogenesis of bone marrow failure in children using single-cell gene expression analysis.

    Grant number:22K07819  2022.4 - 2025.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Scientific Research (C)

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  5. 横紋筋肉腫メタボローム解析(尿/組織)による診断マーカーとオンコメタボライト探索

    Grant number:22K07911  2022.4 - 2025.3

    日本学術振興会  科学研究費助成事業  基盤研究(C)

    住田 亙, 城田 千代栄, 成田 敦, 牧田 智, 田井中 貴久, 内田 広夫, 高橋 義行, 大澤 毅, 田中 裕次郎, 檜 顕成

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    小児の肉腫のなかで最も頻度が高い横紋筋肉腫を標的とし、その中心代謝に関与する代謝産物(オンコメタボライト)及びその特異的な代謝系を見出すことを目標に、生体内の情報を鋭敏且つ包括的に捉えやすいメタボロミクス(キャピラリー電気泳動質量分析法を中心に)に着目する。第一段階として、尿検体を研究サンプルとして用い、尿中代謝物解析(CE/MS:キャピラリー電気泳動質量分析法を中心にLC/MS: 液体クロマトグラフィー質量分析法)から、代謝物を網羅的に測定し、横紋筋肉腫の中心代謝に特異的な代謝物(オンコメタボライト)の解明を目指す。
    横紋筋肉腫は小児軟部組織腫瘍の中で最も頻度が高い。小児横紋筋肉腫の5年無増悪生存率は高リスク群で30~50%と予後不良で現在の診断治療法だけでは未だ十分な治療効果とは言えず、新たな診断治療法の開発が急務である。最近では質量分析計を用いたメタボロミクスの発展により生体内の代謝情報を鋭敏且つ包括的に捉えることが可能となり、様々な癌種において新たなバイオマーカーや病態の解明が進んでいる。横紋筋肉腫においても代謝変動の解明が病因解明や新規治療法の開発に寄与すると考えられるが、その代謝変動に着目した診断マーカーや創薬は前例がない。本研究課題の核心をなす学術的「問い」は横紋筋肉腫に特徴的にみられる代謝産物(オンコメタボライト)や代謝系を見出すことである。本研究では横紋筋肉種患児尿サンプルでメタボローム解析を行い、疾患貢献度の高い尿中代謝物(バイオマーカー)を抽出、更に病態解明を目指して代謝物パスウエイ解析を行い検証する。
    ①詳細な臨床情報を有する尿検体の回収(神経芽腫患児および健常者)、②診療情報を生かした横紋筋肉腫検査モデル構築と妥当性の検証、③代謝経路などの代謝リプログラミング解析の計3項目を検討予定である。
    尿サンプルについては健常者10サンプルと病理組織が胎児型の横紋筋肉腫19サンプル、さらにより悪性度の高い胞巣型の横紋筋肉腫21サンプルを収集し、尿中代謝物を測定した。LC/MS: 液体クロマトグラフィー質量分析法とCE/MS: キャピラリー電気泳動質量分析法で健常者と横紋筋肉腫(胎児型と胞巣型を含む)の尿中代謝物を測定・比較し、横紋筋肉腫群で37代謝物が有意に上昇ないし減少していることが判明した。
    現在、横紋筋肉腫にて有意に上昇している代謝経路などの解析も検証中である。
    ①診療情報を生かした横紋筋肉腫検査モデル構築と妥当性の検証、②代謝経路などの解析の計2項目を中心に研究を行う。引き続き健常者と横紋筋肉腫患者を判別することを尿中代謝物で評価・検証する必要があると考えて いる。
    本研究では今後もLC/MS: 液体クロマトグラフィー質量分析法とCE/MS:キャピラリー電気泳動質量分析法で関連する代謝物を検討する。

  6. CAR-T細胞機能を向上させるCAR構造の最適化

    Grant number:22K07298  2022.4 - 2024.3

    日本学術振興会  科学研究費助成事業  基盤研究(C)

    西尾 信博, 高橋 義行

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    CARの構造は1)抗原認識部位、2)スペーサー、3)共刺激分子、4)細胞内シグナル伝達ドメインにより成り立っている。共刺激分子の違いにより、サイトカイン産生能、増殖能、あるいは体内持続性が異なることが知られているが、共刺激分子以外の構成成分がCAR-T細胞の機能に与える影響は未だ不明である。本研究では、linker部分とspacer部分がCAR-T細胞の機能に与える影響を明らかにし、最適化することでより効果の高いCAR-T細胞を開発する。
    本研究では、CARの構成成分のうち、scFv部分の中のVHとVLをつなぐlinker部分の長さとspacer部分の長さに着目して、CARを改変し、その機能解析を実施する。 ベースとなるCARはGD2を標的としたGD2.CARを用い、遺伝子導入には代表者らが開発したpiggyBacトランスポゾン法を用いる。 異なる抗原認識部位の配列情報とCD28、4-1BBなどの異なる共刺激分子をもつGD2.CARに、さらにlinkerとspacerを改変した複数のGD2.CARコンストラクトを作成した。上記のGD2.CARを、申請者が所有するCD19.CARトランスポゾンベクター(pIRII-CAR.CD19.28.ζ)のCAR配列と入れ替え(pIRII-CAR.GD2.28.ζまたはpIRII-CAR.GD2.4- 1BB.ζ)、GD2.CARトランスポゾンベクターを作成した。GD2CAR-T細胞製造のために培養条件を最適化した。末梢血リンパ球に、ヌクレオフェクション装置を用いて、GD2.CARトランスポゾンベクターとpiggyBac遺伝子転位酵素ベクターを導入し、自家feeder細胞とインターロイキン(IL)-7およびIL-15を添加した培地中で共培養したのちに増殖刺激を加え、GD2CAR-T細胞を作成した。フローサイト メトリーを用いてT細胞上のGD2.CAR発現率、CD45RA/CCR7細胞比率等を測定した。さらにCAR-T細胞と神経芽腫細胞株をサイトカイン無添加・10%血清存在下で共培養し、GD2.CAR-T細胞の抗原特異性、細胞傷害活性、細胞表面exhaustionマーカー、CAR-T細胞のpersistencyなどを検討した。その結果をもとに免疫不全マウスを用いたin vivo実験を実施した。

  7. Development of a novel chimeric antigen receptor gene-modified T cell therapy targeting GD2 for neuroblastoma

    Grant number:19K08273  2019.4 - 2022.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Scientific Research (C)

    Nishio Nobuhiro

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    In this study, in order to develop a new treatment for neuroblastoma, the most common solid tumor among pediatric cancers other than brain tumors, with fewer side effects, we generated a new chimeric antigen receptor CAR targeting GD2 expressed on the cell surface of neuroblastoma cells and used a piggyBac transposon gene transfer method with low production cost. CAR-T cell therapy for neuroblastoma. Multiple GD2.CAR-T cells could be generated by combining multiple antigen recognition sites and co-stimulatory molecules and optimizing the culture method. Functional analysis was performed, and it was possible to select GD2.CAR-T cells with potent tumor cell injury, low exhaustion, and good cell proliferation.

  8. Functional analysis of novel genes related to aplastic anemia using mouse models.

    Grant number:19K08297  2019.4 - 2022.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Scientific Research (C)

    Atsushi Narita

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    We recurrently identified novel gene X mutations using whole exome analysis in children with aplastic anemia. We hypothesized that mutations in gene X might confer an advantage in myeloproliferation or immune escape in aplastic anemia patients. Using a mutant mouse model of gene X, we examined the functional significance of the novel gene X mutant allele in aplastic anemia. Bone marrow specimens were collected from gene X mutant mice and subjected to colony assay, expression analysis by RNA sequencing, MHC deletion analysis, and bone marrow competitive transplantation experiments. In all results, however, mutations in gene X showed no advantage over controls in myeloproliferation or immune escape.

  9. Molecular basis for KIR ligand-mismatched cord blood transplantation for advanced neuroblastoma

    Grant number:18H02690  2018.4 - 2021.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Scientific Research (B)

    Takahashi Yoshiyuki

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    Grant amount:\13650000 ( Direct Cost: \10500000 、 Indirect Cost:\3150000 )

    We were able to establish a Patient-derived Tumor Xenograft (PDX) model using tumors derived from neuroblastoma patients, and established PDX libraries in 5 cases from 18 neuroblastoma patients. Our established PDX model is used in the multicenter prospective clinical study (JN-H-20) nationwide.

  10. Comprehensive gene analysis of clonal hematopoiesis in childhood aplastic anemia

    Grant number:17K16248  2017.4 - 2019.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Young Scientists (B)

    Atsushi Narita, TAKAHASHI Yoshiyuki, MURAMATSU Hideki, OKUNO Yusuke, NAKATOCHI Masahiro

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    We retrospectively studied 57 children with Aplastic anemia (AA) between 1992 and 2010. During the follow‐up, five patients developed clinical PNH, in whom somatic PIGA mutations were detected by targeted sequencing. The 10‐year probability of clinical PNH development was 10.2% (95% CI, 3.6%-20.7%). Furthermore, the detection of minor PNH clones by flow cytometry at AA diagnosis was a risk factor for the subsequent development of clinical PNH.
    We performed whole-exome sequencing (WES) of paired bone marrow-germline reference samples in 41 patients. WES detected reccurent gene X mutations as a novel mutational target. Gene X mutation may be associated the pahophisiology of AA.

  11. Mechanisms of carcinogenesis through deviation from normal development

    Grant number:15K15079  2015.4 - 2017.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Challenging Exploratory Research

    Kadomatsu Kenji, Kiyonari Shinichi, Takeuchi Ichiro, Takahashi Yoshiyuki

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    Why does tumor develop without driver gene mutations in pediatric cancers? Its reason remains elusive although it is speculated that epigenetic regulations are involved. To address this question, we developed a new method, i.e., tumor sphere culture from very early stages of carcinogenesis. We found that cancer has developed as early as in mid-gestation period in the neuroblastoma model TH-MYCN mice. Furthermore, we found that epigenetic regulations are indeed critical for neuroblastoma development, and that EZH2 is a molecular target for its treatment.

  12. Development of Epstein-Barr virus (EBV) specific cytotoxic T lymphocytes (CTL) therapy for EBV-LPD after hematopoietic stem cell transplantation

    Grant number:26461580  2014.4 - 2017.3

    Japan Society for the Promotion of Science  Grants-in-Aid for Scientific Research  Grant-in-Aid for Scientific Research (C)

    Takahashi Yoshiyuki, KIMURA Hiroshi, MIZUNO Masaaki, LEEN Ann, SAJI Hiroo

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    We analyzed the relation of EBV reactivation and Anti-thymocyte globulin (ATG) blood concentration in 90 cases and revealed that EBV reactivation was significantly more in high ATG concentration group. We could successfully developed EBV specific CTLs from 3rd party donors using not only HLA A2402/A0201 peptides but also HLA-A11 restricted peptides. We started the phase 1 clinical trial of EBV specific CTL therapy from the 3rd party donors.

  13. 小児難治性急性骨髄性白血病に対するアロ反応性NK細胞による抗腫瘍メカニズムの解明

    2008

    科学研究費補助金  基盤研究(C),課題番号:20591252

    高橋 義行

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