2022/07/19 更新

写真a

ヤマモト ヒロユキ
山本 啓之
YAMAMOTO Hiroyuki
所属
医学部附属病院 小児科 助教
大学院担当
大学院医学系研究科
職名
助教
外部リンク

学位 1

  1. 博士(医学) ( 2016年1月   名古屋大学 ) 

研究分野 1

  1. ライフサイエンス / 胎児医学、小児成育学  / 小児神経 てんかん 神経生理

経歴 9

  1. 名古屋大学   小児科   助教

    2019年9月 - 現在

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  2. 名古屋大学   小児科   医員

    2016年4月 - 2019年8月

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  3. 北海道大学   小児科   研究員

    2015年4月 - 2016年3月

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  4. 愛知県青い鳥医療福祉センター   小児科

    2014年4月 - 2015年3月

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  5. 名古屋大学   小児科

    2010年10月 - 2014年3月

  6. 愛知県厚生連安城更生病院   小児科

    2009年9月 - 2010年9月

  7. 名古屋大学   小児科

    2009年4月 - 2009年9月

  8. 愛知県心身障害者コロニー中央病院   新生児科

    2008年4月 - 2009年3月

  9. 名古屋掖済会病院   初期研修医

    2004年4月 - 2006年3月

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学歴 2

  1. 名古屋大学大学院医学系研究科   健康社会医学専攻

    2010年4月 - 2014年3月

  2. 名古屋大学   医学部   医学科

    1998年4月 - 2004年3月

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    国名: 日本国

所属学協会 7

  1. 日本小児科学会

  2. 日本臨床神経生理学会   代議員

  3. 日本てんかん学会

  4. 日本小児神経学会

  5. 日本臨床神経生理学会

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  6. 日本小児科学会

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  7. 日本小児神経学会

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委員歴 2

  1. 日本てんかん学会   てんかん学教育委員  

    2021年10月   

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    団体区分:学協会

  2. 日本てんかん学会   法的問題検討委員会  

    2021年10月   

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    団体区分:学協会

 

論文 40

  1. Acute encephalopathy with biphasic seizures and late reduced diffusion: Predictive EEG findings

    Ohno Atsuko, Okumura Akihisa, Fukasawa Tatsuya, Nakata Tomohiko, Suzuki Motomasa, Tanaka Masaharu, Okai Yu, Ito Yuji, Yamamoto Hiroyuki, Tsuji Takeshi, Kidokoro Hiroyuki, Saitoh Shinji, Natsume Jun

    BRAIN & DEVELOPMENT   44 巻 ( 3 ) 頁: 221 - 228   2022年3月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    Background: Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is a common type of acute encephalopathy in Japan; the condition is clinically characterized by prolonged seizures as the initial neurological symptom, followed by late seizures 4–6 days later. It is difficult to differentiate AESD from prolonged febrile seizures (PFSs). Here, we explored the use of electroencephalography to differentiate AESD from PFSs. Methods: We studied the electroencephalograms (EEGs) of children <6 years of age diagnosed with AESD or PFSs; all EEGs were recorded within 48 h of seizure onset (i.e., before the late seizures of AESD). Two pediatric neurologists evaluated all EEGs, focusing on the basic rhythm, slowing during wakefulness/arousal by stimuli, spindles, fast waves, and slowing during sleep. Results: The EEGs of 14 children with AESD and 31 children with PFSs were evaluated. Spindles were more commonly reduced or absent in children with AESD than in those with PFSs (71% vs. 31%, p = 0.021). Fast waves were also more commonly reduced or absent in children with AESD (21% vs. 0%, p = 0.030). The rates of all types of slowing did not differ between children with AESD and those with PFSs, but continuous or frequent slowing during sleep was more common in the former (50% vs. 17%, p = 0.035). Conclusions: EEG findings may usefully differentiate AESD from PFSs. Reduced or absent spindles/fast waves and continuous or frequent slowing during sleep are suggestive of AESD in children with prolonged seizures associated with fever.

    DOI: 10.1016/j.braindev.2021.11.003

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  2. Involvement of brain structures in childhood epilepsy with centrotemporal spikes

    Ito Yuji, Maki Yuki, Okai Yu, Kidokoro Hiroyuki, Bagarinao Epifanio, Takeuchi Tomoya, Ohno Atsuko, Nakata Tomohiko, Ishihara Naoko, Okumura Akihisa, Yamamoto Hiroyuki, Maesawa Satoshi, Natsume Jun

    PEDIATRICS INTERNATIONAL   64 巻 ( 1 ) 頁: e15001   2022年1月

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    記述言語:日本語   出版者・発行元:Pediatrics international : official journal of the Japan Pediatric Society  

    BACKGROUND: We aimed to investigate electroencephalography (EEG)-functional magnetic resonance imaging (fMRI) findings to elucidate the interictal epileptiform discharge (IED)-related functional alterations in deep brain structures and the neocortex in childhood epilepsy with centrotemporal spikes (CECTS). METHODS: Ten children with CECTS (median age 8.2 years), referred to our hospital within a year of onset, were eligible for inclusion. They underwent EEG-fMRI recording during sleep. Llongitudinal evaluations, including medical examinations, intelligence tests, and questionnaires about developmental disabilities, were performed. The initial evaluation was performed at the same time as the EEG-fMRI, and the second evaluation was performed over 2 years after the initial evaluation. RESULTS: Three children were unable to maintain sleep during the EEG-fMRI recording, and the remaining seven children were eligible for further assessment. All patients showed unilateral-dominant centrotemporal spikes during scans. One patient had only positive hemodynamic responses, while the others had both positive and negative hemodynamic responses. All patients showed IED-related hemodynamic responses in the bilateral neocortex. For deep brain structures, IED-related hemodynamic responses were observed in the cingulate gyrus (n = 4), basal ganglia (n = 3), thalamus (n = 2), and default mode network (n = 1). Seizure frequencies at the second evaluation were infrequent or absent, and the longitudinal results of intelligence tests and questionnaires were within normal ranges. CONCLUSIONS: We demonstrated that IEDs affect broad brain areas, including deep brain structures such as the cingulate gyrus, basal ganglia, and thalamus. Deep brain structures may play an important role in the pathophysiology of CECTS.

    DOI: 10.1111/ped.15001

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

    Suzuki Takeshi, Natsume Jun, Kumai Sumire, Maki Yuki, Yamamoto Hiroyuki, Numoto Shingo, Narahara Sho, Kubota Tetsuo, Tsuji Takeshi, Kato Toru, Yamada Keitaro, Maruyama Koichi, Okumura Akihisa, Takahashi Yoshiyuki, Kidokoro Hiroyuki

    EPILEPSY & BEHAVIOR   125 巻   頁: 108397   2021年12月

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    記述言語:日本語   出版者・発行元:Epilepsy and Behavior  

    Purpose: This multicenter study examined the effectiveness and tolerability of lacosamide (LCM) for children and young adults with epilepsy, particularly in patients who had previously been treated with other sodium channel blockers (SCBs) and the difference in effectiveness and tolerability when using other concomitant SCBs. Methods: We retrospectively studied the clinical information of patients aged <30 years given LCM to treat epilepsy. The effectiveness and adverse events (AEs) of LCM and the other SCBs were investigated. Factors related to the effectiveness and AEs of LCM, such as the number of antiepileptic drugs (AEDs) tried before LCM and concomitantly used SCBs, were also studied. Results: We enrolled 112 patients (median age = 11 years). One year after starting LCM, 29% of the patients were seizure free, and 50% had a ≥50% seizure reduction. Of the patients, 17% experienced AEs, the most common being somnolence. A ≥50% seizure reduction was observed for LCM in 30% of patients in whom other SCBs had not been effective. Lacosamide produced a ≥50% seizure reduction in 35% of the patients taking one concomitant SCB. By contrast, no patients had ≥50% seizure reduction, and 33% developed AEs, when LCM was administered concomitantly with two SCBs. Conclusions: Lacosamide was effective in 30% of children and young adults in whom other SCBs had not been effective. The effectiveness of LCM may differ from that of other SCBs, and it is worth trying in patients with epilepsy resistant to other AEDs.

    DOI: 10.1016/j.yebeh.2021.108397

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  4. Trajectory of the incidence of brushes on preterm electroencephalogram and its association with neurodevelopment in extremely low birth weight infants

    Maeda Takashi, Kidokoro Hiroyuki, Tachibana Takashi, Shiraki Anna, Yamamoto Hiroyuki, Nakata Tomohiko, Fukasawa Tatsuya, Kubota Tetsuo, Sato Yoshiaki, Kato Toru, Natsume Jun, Okumura Akihisa, Hayakawa Masahiro

    BRAIN & DEVELOPMENT   43 巻 ( 10 ) 頁: 979 - 987   2021年11月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    Background: Brush or delta brush is a well-known characteristic waveform in preterm electroencephalograms. However, the longitudinal trajectory of brushes and its association with neurodevelopment remain uncertain. Methods: We analyzed the longitudinal incidence of brushes in 36 extremely low birth weight infants without severe brain lesions and its association with neurodevelopment and white matter abnormality. Conventional eight-channel electroencephalograms were recorded at 30, 32, 36, and 40 postmenstrual weeks (PMW). Incidence of brushes was calculated as the sum of brushes from each channel separated by active sleep and quiet sleep. A developmental delay was defined as a developmental quotient of <85 assessed at corrected age of 18 months. White matter abnormalities were evaluated with term-equivalent magnetic resonance imaging. Results: The median incidence of brushes (per minute) in 36 infants at PMW 30, 32, 36, and 40 was 16.4, 20.4, 22.5, and 1.8 during active sleep and 7.5, 10.3, 11.5, and 1.7 during quiet sleep, respectively. Among the 36 infants, 14 infants were diagnosed with developmental delay. Longitudinal trajectories of the incidence of brushes were different between the normal and the delayed development groups. Brushes were observed most frequently at 36 PMW in the delayed development group. The incidence of brushes at 36 PMW was significantly correlated with the severity of white matter abnormalities and negatively correlated with the developmental quotient. Conclusion: The incidence of brushes at 36 PMW can be a unique predictor of early neurodevelopment in extremely low birth weight infants without severe brain lesions.

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  5. Repetitive sleep starts: An important differential diagnosis of infantile spasms 国際誌

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

    EPILEPSY & BEHAVIOR   121 巻 ( Pt A ) 頁: 108075 - 108075   2021年8月

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    記述言語:日本語   掲載種別:研究論文(学術雑誌)   出版者・発行元:Epilepsy and Behavior  

    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.

    DOI: 10.1016/j.yebeh.2021.108075

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

    Kawaguchi Masahiro, Kidokoro Hiroyuki, Ito Rintaro, Shiraki Anna, Suzuki Takeshi, Maki Yuki, Tanaka Masaharu, Sakaguchi Yoko, Yamamoto Hiroyuki, Takahashi Yosiyuki, Naganawa Shinji, Natsume Jun

    MAGNETIC RESONANCE IMAGING   79 巻   頁: 38 - 44   2021年6月

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    記述言語:日本語   掲載種別:研究論文(学術雑誌)   出版者・発行元:Magnetic Resonance Imaging  

    The accuracy of brain age estimates from magnetic resonance (MR) images has improved with the advent of deep learning artificial intelligence (AI) models. However, most previous studies on predicting age emphasized aging from childhood to adulthood and old age, and few studies have focused on early brain development in children younger than 2 years of age. Here, we performed brain age estimates based on MR images in children younger than 2 years of age using deep learning. Our AI model, developed with one slice each of raw T1- and T2-weighted images from each subject, estimated brain age with a mean absolute error of 8.2 weeks (1.9 months). The estimates of our AI model were close to those of human specialists. The AI model also estimated the brain age of subjects with a myelination delay as significantly younger than the chronological age. These results indicate that the prediction accuracy of our AI model approached that of human specialists and that our simple method requiring less data and preprocessing facilitates a radiological assessment of brain development, such as monitoring maturational changes in myelination.

    DOI: 10.1016/j.mri.2021.03.004

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  7. Risks of ACTH therapy for West syndrome following BCG vaccination 国際誌

    Maki Yuki, Natsume Jun, Hori Ikumi, Takeuchi Tomoya, Negishi Yutaka, Kubota Tetsuo, Maruyama Koichi, Nakata Tomohiko, Yamamoto Hiroyuki, Tanaka Masaharu, Kawaguchi Masahiro, Suzuki Takeshi, Shiraki Anna, Sawamura Fumi, Kidokoro Hiroyuki

    EPILEPSY & BEHAVIOR   118 巻   頁: 107924 - 107924   2021年5月

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    記述言語:日本語   掲載種別:研究論文(学術雑誌)   出版者・発行元:Epilepsy and Behavior  

    Objective: Bacille de Calmette et Guérin (BCG) is a live vaccine for tuberculosis that is administered to all infants in Japan. Adrenocorticotropic hormone (ACTH) therapy for West syndrome (WS) causes immunosuppression and may result in BCG infection after BCG vaccination. We evaluated the safety of ACTH therapy initiated shortly after BCG vaccination. Methods: We analyzed patients with WS who received ACTH therapy between 2005 and 2018. We evaluated the interval between BCG and ACTH therapy, and the rate of BCG infection during and after ACTH therapy, by retrospective chart review. Results: Seventy-nine patients were included in the analysis. Twenty-three patients received ACTH therapy prior to BCG vaccination. For the remaining 56 patients, the median interval between BCG vaccination and the start of ACTH therapy (BCG–ACTH interval) was 91.5 (range 14–280) days. The BCG–ACTH interval was shorter in patients with unknown than in those with known etiologies. It was <8 weeks in 13 patients (10 with unknown and 3 with known etiologies). The minimum BCG–ACTH interval was 14 days. Six patients with epileptic spasms received BCG vaccinations because physicians did not recognize their seizures. None of the patients developed BCG infection. Conclusion: No patients who received ACTH therapy after BCG, even at an interval of 8 weeks, developed BCG infection. The timing of ACTH therapy initiation should be based on the risk of BCG-related adverse events and the adverse effects of any delay.

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  8. MRI findings in children with congenital cytomegalovirus infection retrospectively diagnosed with dried umbilical cord

    Kidokoro Hiroyuki, Shiraki Anna, Torii Yuka, Tanaka Masaharu, Yamamoto Hiroyuki, Kurahashi Hirokazu, Maruyama Koichi, Okumura Akihisa, Natsume Jun, Ito Yoshinori

    NEURORADIOLOGY   63 巻 ( 5 ) 頁: 761 - 768   2021年5月

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    記述言語:日本語   出版者・発行元:Neuroradiology  

    Purpose: Brain MRI provides important information about suspected congenital CMV infection in neonatally underdiagnosed children. This study aimed to describe MRI findings in children in whom congenital CMV infection was not suspected during the neonatal period and was proven retrospectively. Methods: We enrolled 31 children referred to the pediatric neurology clinic with neurological symptoms who were proven to have congenital CMV infection based on dried umbilical cord samples. Upon diagnosis, MR and CT images were assessed using the van der Knaap scoring system integrated with additional variables. Two investigators independently assessed all images. Results: The age at diagnosis was < 12 months in 14, 12–24 months in 11, and > 24 months in 6 patients. The initial symptom triggering clinic referral was delayed development in 22, seizure in 5, deafness in 3, and hemiplegia in 1 patient. Of the 31 children, 30 had a white matter (WM) abnormality predominant in the deep WM of the parietal lobe (n = 25). Anterior temporal lesions were observed in 21 children. Cortical lesions were observed in 7 children, suggestive of polymicrogyria. No child had cerebellar or brainstem abnormalities. Brain CT was performed in 22 of 31 children, and 11 showed punctate cerebral calcification in the periventricular and/or deep WM. Conclusion: Patients with congenital CMV infection with delayed neurological symptoms show a relatively uniform pattern of parietal-dominant multifocal WM lesions and anterior temporal lesions, with or without polymicrogyria.

    DOI: 10.1007/s00234-020-02603-9

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  9. Shuffling babies and autism spectrum disorder 国際誌

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

    BRAIN & DEVELOPMENT   43 巻 ( 2 ) 頁: 181 - 185   2021年2月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    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.

    DOI: 10.1016/j.braindev.2020.08.007

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  10. Lenticular nuclei to thalamic ratio on PET is useful for diagnosis of GLUT1 deficiency syndrome

    Natsume Jun, Ishihara Naoko, Azuma Yoshiteru, Nakata Tomohiko, Takeuchi Tomoya, Tanaka Masaharu, Sakaguchi Yoko, Okai Yu, Ito Yuji, Yamamoto Hiroyuki, Ohno Atsuko, Kidokoro Hiroyuki, Hattori Ayako, Nabatame Shin, Kato Katsuhiko

    BRAIN & DEVELOPMENT   43 巻 ( 1 ) 頁: 69 - 77   2021年1月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    Purpose: To establish an objective method of [18F]-fluorodeoxyglucose positron emission tomography (FDG-PET) that can assist in the diagnosis of glucose transporter 1 deficiency syndrome (GLUT1-DS). Methods: FDG-PET was performed in 8 patients with a mean age of 12.5 years (range, 2–22 years) with GLUT1-DS. Their PET findings were compared with those of 45 controls with a mean age of 11.2 years (range, 2–21 years) by statistical parametric mapping (SPM12, Welcome Neurological Institute). The controls had epilepsy of unknown etiology and normal MRI findings. The age-adjusted ratios of mean radioactivities in regions of interest (ROIs) of bilateral lenticular nuclei, thalami, and the whole cerebral cortex were also measured. The sensitivities and specificities of the ratios for the differential diagnosis of GLUT1-DS were also determined. Results: SPM showed significantly decreased uptake in bilateral thalami and increased uptake in bilateral lenticular nuclei in patients with GLUT1-DS. There were no areas in the cerebral cortex with significant differences between patients and controls. On ROI analysis, by setting the cut-off value of the age-adjusted lenticular nuclei/thalami radioactivity ratio to 1.54, patients with GLUT1-DS were differentiated from controls with sensitivity of 1.00 and specificity of 0.98. Conclusion: The age-adjusted lenticular nuclei/thalami radioactivity ratio on PET can distinguish patients with GLUT1-DS from patients with epilepsy of unknown etiology with high sensitivity and specificity. It is important to pay attention to the metabolism of the lenticular nuclei and thalami on PET for the diagnosis of GLUT1-DS.

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  11. ホスフェニトインが有効であった新生児発作の1例

    橋本 実沙, 神澤 孝洋, 山本 啓之, 加藤 英子, 家田 訓子

    日本周産期・新生児医学会雑誌   57 巻 ( 1 ) 頁: 124 - 128   2021年

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    記述言語:日本語   出版者・発行元:一般社団法人 日本周産期・新生児医学会  

    <p> フェノバルビタール(以下PB)及びミダゾラム(以下MDL)に抵抗性の新生児発作の治療について十分なエビデンスは未だ得られていない.新生児発作にホスフェニトイン(以下fos-PHT)が有効であった1例を報告する.症例は,在胎40週5日に胎児機能不全のため吸引分娩にて出生した.Apgar Score1点(1分),4点(5分),5点(10分)で,新生児仮死が疑われNICUに入院した.生後32時間よりamplitude-integrated EEG(以下aEEG)波形は鋸歯状パターンを呈し,脳波波形では律動的に反復する同一波形を認めた.左上肢の間代性けいれんも出現し新生児発作と診断した.PB及びMDL投与で発作は持続し,fos-PHT投与で発作は消失した.低血圧や徐脈などの副作用は認めなかった.両親にfos-PHTが新生児に対し適応外使用である旨を説明し同意を得た.本症例では新生児発作にfos-PHTが有効で,安全に使用することができた.</p>

    DOI: 10.34456/jjspnm.57.1_124

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  12. Change of White Matter Integrity in Children With Hematopoietic Stem Cell Transplantation

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

    PEDIATRIC NEUROLOGY   111 巻   頁: 78 - 84   2020年10月

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    記述言語:日本語   出版者・発行元:Pediatric Neurology  

    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 significantly high 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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  13. High-amplitude fast activity in EEG: An early diagnostic marker in children with beta-propeller protein-associated neurodegeneration (BPAN)

    Kidokoro Hiroyuki, Yamamoto Hiroyuki, Kubota Tetsuo, Motobayashi Mitsuo, Miyamoto Yusaku, Nakata Tomohiko, Takano Kyoko, Shiba Naoko, Okai Yu, Tanaka Masaharu, Sakaguchi Yoko, Maki Yuki, Kawaguchi Masahiro, Suzuki Takeshi, Muramatsu Kazuhiro, Natsume Jun

    CLINICAL NEUROPHYSIOLOGY   131 巻 ( 9 ) 頁: 2100 - 2104   2020年9月

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    記述言語:日本語   出版者・発行元:Clinical Neurophysiology  

    Objective: The early diagnosis of beta-propeller protein-associated neurodegeneration (BPAN) before distinct brain magnetic resonance imaging (MRI) findings of iron deposition occur remains challenging. This study examined whether children with BPAN have characteristic high-amplitude (>50 μV) fast activity (HAFA) on electroencephalography (EEG). Methods: We conducted a retrospective analysis of EEG performed during childhood in five patients with BPAN. We also examined 143 EEGs from 59 patients with different etiologies, including epilepsy (n = 33), acute encephalopathy (n = 6), neurodevelopmental disorders (n = 5), non-epileptic events (n = 4), and others (n = 11). Trained electroencephalographers reviewed all of the EEGs. When excessive fast activity was observed, the amplitude, frequency, and locality were assessed. Results: All five patients with BPAN underwent initial EEGs at 12–21 months old, and diffuse continuous HAFA (range 20–50 Hz) was observed on both awake and sleep EEGs. In the awake records, there was no clear posterior dominant rhythm in 4 of the 5 patients. Although 28% of the 143 EEGs had continuous excessive fast activity, mainly in the sleep records, only two (1.4%) exhibited HAFA when asleep, and their awake EEGs had clear posterior dominant rhythm. Conclusions: The EEGs of children with BPAN showed diffuse HAFA continuously when both awake and asleep, which is uncommon in children with other etiologies. Significance: This study provides an important clue for the early diagnosis of BPAN.

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  14. Transient cortical diffusion restriction in children immediately after prolonged febrile seizures

    Suzuki Takeshi, Kidokoro Hiroyuki, Kubota Tetsuo, Fukasawa Tatsuya, Suzui Ryosuke, Tsuji Takeshi, Kato Toru, Yamamoto Hiroyuki, Ohno Atsuko, Nakata Tomohiko, Saitoh Shinji, Okumura Akihisa, Natsume Jun

    EUROPEAN JOURNAL OF PAEDIATRIC NEUROLOGY   27 巻   頁: 30 - 36   2020年7月

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    記述言語:日本語   出版者・発行元:European Journal of Paediatric Neurology  

    Aim: Little is known about acute febrile status epilepticus-induced injury of extrahippocampal structures. To clarify the presence and clinical significance of acute extrahippocampal injuries, we performed diffusion-weighted imaging (DWI) in children immediately after prolonged febrile seizure (PFS). Method: We performed a retrospective cohort study in children younger than 6 years old who visited one of two hospitals due to PFSs between January 2013 and October 2018. PFS was defined as a febrile seizure that persisted for 15 min or longer. We collected brain DWI data within 6 h of the end of PFS. When the initial DWI detected an abnormality, a follow-up DWI was performed a few days later. Results: The study population consisted of 101 patients with PFSs. DWI was performed within 6 h in 51 patients, while the remaining 50 patients did not undergo imaging because of good recovery of consciousness. Restricted cortical diffusion was evident in 9 (18%) patients on initial DWI. All of them underwent DWI within 100 min after PFS. Restricted cortical diffusion was associated with male sex, asymmetrical PFS symptoms, and a shorter duration between the end of the seizure and DWI, but was not associated with seizure duration. All cortical abnormalities had resolved on follow-up DWI of these patients within 72 h after the initial imaging, but ipsilateral hippocampal hyperintensity appeared in one patient. All 9 patients with restricted cortical diffusion were finally diagnosed with PFS and discharged without sequelae. Conclusions: Some children with PFSs exhibit transient restricted diffusion in the regional cortex on DWI performed immediately after the end of PFS. These transient diffusion changes were not associated with unfavorable epileptic sequelae or neuroimaging in the short-term.

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  15. Subsecond EEG-fMRI analysis for presurgical evaluation in focal epilepsy 国際誌

    Ito Yuji, Maesawa Satoshi, Bagarinao Epifanio, Okai Yu, Nakatsubo Daisuke, Yamamoto Hiroyuki, Kidokoro Hiroyuki, Usui Naotaka, Natsume Jun, Hoshiyama Minoru, Wakabayashi Toshihiko, Sobue Gen, Ozaki Norio

    JOURNAL OF NEUROSURGERY   134 巻 ( 4 ) 頁: 1027 - 1036   2020年4月

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    記述言語:日本語   掲載種別:研究論文(学術雑誌)   出版者・発行元:Journal of Neurosurgery  

    OBJECTIVE The authors recently reported a novel subsecond analysis method of analyzing EEG–functional MRI (fMRI) to improve the detection rate of epileptic focus. This study aims to validate the utility of this method for presurgical evaluation in pharmacoresistant focal epilepsy. METHODS Among 13 patients with focal epilepsy undergoing presurgical examinations including simultaneous EEG-fMRI at 3T, 11 patients had interictal epileptiform discharges (IEDs) during fMRI. The authors used the sequence of topographic maps during the IEDs as a reference to obtain subsecond fMRI activation maps with the same temporal resolution as the EEG data, and constructed “spike-and-slow-wave-activation-summary” (SSWAS) maps that showed the activation frequency of voxels during IEDs. Clusters were defined by thresholding the SSWAS maps (voxel value > 10), and those containing voxels with the top 3 highest activation frequencies were considered significant. Significant hemodynamic responses using conventional event-related (ER) analysis and SSWAS maps were compared with the resection areas and surgical outcomes at 1 year after surgery. RESULTS Using ER analysis, 4 (36%) of 11 patients had significant hemodynamic responses. One of 4 patients had significant hemodynamic responses in the resection area and good surgical outcome. Using SSWAS maps, 10 (91%) of 11 patients had significant hemodynamic responses. Six of 10 patients had significant hemodynamic responses in the resection area, and 5 of the 6 patients achieved good surgical outcomes. The remaining 4 patients had significant hemodynamic responses distant from the resection area, and only 1 of the 4 patients achieved good surgical outcomes. The sensitivity, specificity, positive predictive value, and negative predictive value of SSWAS maps were 83.3%, 75.0%, 83.3%, and 75.0%, respectively. CONCLUSIONS This study demonstrated the clinical utility of SSWAS maps for presurgical evaluation of pharmacoresistant focal epilepsy. The findings indicated that subsecond EEG-fMRI analysis may help surgeons choose the resection areas that could lead to good surgical outcomes.

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  16. y Distributed source analysis of magnetoencephalography using a volume head model combined with statistical methods improves focus diagnosis in epilepsy surgery

    Ishizaki Tomotaka, Maesawa Satoshi, Nakatsubo Daisuke, Yamamoto Hiroyuki, Takai Sou, Shibata Masashi, Kato Sachiko, Natsume Jun, Hoshiyama Minoru, Wakabayashi Toshihiko

    SCIENTIFIC REPORTS   10 巻 ( 1 ) 頁: 5263   2020年3月

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    記述言語:日本語   出版者・発行元:Scientific Reports  

    Deep-seated epileptic focus estimation using magnetoencephalography is challenging because of its low signal-to-noise ratio and the ambiguity of current sources estimated by interictal epileptiform discharge (IED). We developed a distributed source (DS) analysis method using a volume head model as the source space of the forward model and standardized low-resolution brain electromagnetic tomography combined with statistical methods (permutation tests between IEDs and baselines and false discovery rate between voxels to reduce variation). We aimed to evaluate the efficacy of the combined DS (cDS) analysis in surgical cases. In total, 19 surgical cases with adult and pediatric focal epilepsy were evaluated. Both cDS and equivalent current dipole (ECD) analyses were performed in all cases. The concordance rates of the two methods with surgically identified epileptic foci were calculated and compared with surgical outcomes. Concordance rates from the cDS analysis were significantly higher than those from the ECD analysis (68.4% vs. 26.3%), especially in cases with deep-seated lesions, such as in the interhemispheric, fronto-temporal base, and mesial temporal structures (81.8% vs. 9.1%). Furthermore, the concordance rate correlated well with surgical outcomes. In conclusion, cDS analysis has better diagnostic performance in focal epilepsy, especially with deep-seated epileptic focus, and potentially leads to good surgical outcomes.

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  17. Pseudo-sawtooth pattern on amplitude-integrated electroencephalography in neonatal hypoxic-ischemic encephalopathy

    Tanaka Masaharu, Kidokoro Hiroyuki, Kubota Tetsuo, Fukasawa Tatsuya, Okai Yu, Sakaguchi Yoko, Ito Yuji, Yamamoto Hiroyuki, Ohno Atsuko, Nakata Tomohiko, Negoro Tamiko, Okumura Akihisa, Kato Toru, Watanabe Kazuyoshi, Takahashi Yoshiyuki, Natsume Jun

    PEDIATRIC RESEARCH   87 巻 ( 3 ) 頁: 529 - 535   2020年2月

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    記述言語:日本語   出版者・発行元:Pediatric Research  

    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.

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  18. Novel biallelic FA2H mutations in a Japanese boy with fatty acid hydroxylase-associated neurodegeneration

    Kawaguchi Masahiro, Sassa Takayuki, Kidokoro Hiroyuki, Nakata Tomohiko, Kato Kohji, Muramatsu Hideki, Okuno Yusuke, Yamamoto Hiroyuki, Kaname Tadashi, Kihara Akio, Natsume Jun

    BRAIN & DEVELOPMENT   42 巻 ( 2 ) 頁: 217 - 221   2020年2月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    FA2H encodes fatty acid 2-hydroxylase, which plays a significant role in maintaining the neuronal myelin sheath. Previous reports have revealed that a FA2H mutation leads to spastic paraplegia, leukodystrophy, and neurodegeneration with brain iron accumulation, collectively referred to as fatty acid hydroxylase-associated neurodegeneration (FAHN). The disease severity of FAHN varies among individual patients and may be explained by the enzyme activity of FA2H mutant proteins. Here we report a 10-year-old Japanese boy with FAHN having novel heterozygous mutations in FA2H. The patient presented with a spastic gait since the age of 5 years and was unable to walk without a cane by the time he was 8 years old. Brain MRI demonstrated a partial thinning of the corpus callosum, slight reduction of cerebellar volume, and posterior dominant periventricular leukodystrophy. Whole exome sequencing revealed two novel missense mutations in FA2H with compound heterozygous inheritance (NM_024306, p.Val149Leu, and p.His260Gln mutations). The enzyme activities of the p.Val149Leu and p.His260Gln variants were 60%–80% and almost 0%, respectively. Our cell-based enzyme assay demonstrated partial functionality for one of the variants, indicating a milder phenotype. However, considered along with previous reports, there was no definite relationship between the disease severity and residual enzyme activity measured using a similar method. Further research is needed to precisely predict the phenotypic severity of this disorder.

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  19. Attitudes of school teachers toward epilepsy in Nagoya, Japan

    Okumura Akihisa, Saitoh Shinji, Natsume Jun, Yamamoto Hiroyuki, Kurahashi Hirokazu, Numoto Shingo

    EPILEPSY & BEHAVIOR   103 巻 ( Pt A ) 頁: 106359   2020年2月

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    記述言語:日本語   出版者・発行元:Epilepsy and Behavior  

    We conducted a questionnaire survey to examine the knowledge about and attitudes toward epilepsy among school teachers in an urban area in Japan. The questionnaire consisted of six questions: two questions about contact with children with epilepsy, one about knowledge about emergency care, and three about attitudes toward the participation of children with epilepsy in school events. The respondents answered each question using a five-point Likert scale: strongly agree, agree, undecided, disagree, or strongly disagree. A total of 713 teachers completed the questionnaire. More than one-third of the teachers had taught children with epilepsy, and more than half had witnessed a seizure. Although 36% thought that one should “put something in the mouth during a seizure”, fewer believed in restricting swimming, out-of-school activities, and sports. Special school teachers were less likely to suggest putting something in the mouth during a seizure and restrict swimming than were normal school teachers. The answers did not differ according to teacher age or contact with children with epilepsy. Contact with children with epilepsy was relatively common among school teachers, whereas knowledge about epilepsy was insufficient. Negative attitudes toward epilepsy were uncommon. Special school teachers knew more about epilepsy than normal school teachers.

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  20. 急性脳炎脳症に対する脳波モニタリングを用いた治療介入

    山本 啓之

    脳と発達   52 巻 ( 4 ) 頁: 246 - 248   2020年

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    記述言語:日本語   出版者・発行元:一般社団法人 日本小児神経学会  

    <p>脳波検査は古くから人の脳機能を把握する検査として行われている. 急性脳症における知見も積み重ねられてはいるが, 近年その診断には頭部MRIの存在感が高まっている. しかし, 脳波は脳機能をリアルタイムに, また持続的に把握できる検査法であり, 急性脳症・脳炎では必ず考慮されるべき検査法である. 急性脳症では頭部MRIよりも早く異常所見を呈することがあり, 脳波検査により早期に診断できる可能性がある. また, 持続脳波モニタリングを行うことで潜在性の発作を検出することができ, 発作の早期治療介入により予後を改善させることが期待される. 血圧, 心電図, 酸素飽和度のように生体情報モニターとして脳波が使用されていくことを望む.</p>

    DOI: 10.11251/ojjscn.52.246

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  21. 序論

    山本 啓之, 前垣 義弘

    脳と発達   52 巻 ( 4 ) 頁: 238 - 239   2020年

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    記述言語:日本語   出版者・発行元:一般社団法人 日本小児神経学会  

    DOI: 10.11251/ojjscn.52.238

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  22. Serial evaluation of myelin oligodendrocyte glycoprotein antibody in a child with multiphasic acute disseminated encephalomyelitis

    Kumai S, Yamamoto H, Nakata T, Kidokoro H, Fujiura N, Shibata M, Kaneko K, Takahashi T, Natsume J

    No To Hattatsu   52 巻 ( 6 ) 頁: 414 - 418   2020年

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    記述言語:日本語   出版者・発行元:No To Hattatsu  

    Myelin oligodendrocyte glycoprotein antibody (MOG-Ab) is identified in some patients with ADEM. Whether serial change of MOGAb titer is useful for deciding on the therapeutic strategy for multiphasic ADEM has not been clarified. We report the case of multiphasic ADEM who was treated with intravenous immunoglobulin (IVIG) therapy and whose MOG-Ab was serially evaluated. The patient is 9-yearold boy. He showed acute onset of headache and delirium. T2-weighted and fluid attenuated inversion recovery (FLAIR) magnetic resonance imaging (MRI) showed multiple hyperintense lesions in bilateral subcortical and cortical areas, and we diagnosed the patient with ADEM. Although symptoms and MRI lesions were resolved by steroid pulse therapy, he experienced three time relapses characterized by headache and abnormal behavior as well as new MRI lesions during tapering of the therapy. We started monthly IVIG from 5 months after onset, and no relapse of ADEM was encountered. MOG-Ab titer ranged from 4,096 to 2,048 before IVIG therapy. After starting IVIG, MOG-Ab titer gradually decreased. We were able to decrease and discontinue oral steroids. After steroid discontinuation, MOG-Ab titer remained low. We started to decrease the dose of IVIG at 1 year and 3 months after the onset of ADEM and terminated IVIG at 1 year and 9 months after onset by checking low titer of MOG-Ab. Our results suggest that serial evaluations of MOG-Ab may contribute to the monitoring of disease activity and decision-making on drug tapering and termination for multiphasic ADEM. The utility of serial evaluations should be validated in studies of larger numbers of patients.

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  23. 小児の急性脳症,熱性けいれん重積における脳波

    夏目 淳, 大野 敦子, 山本 啓之, 城所 博之, 沼口 敦

    Journal of Japan Society of Neurological Emergencies & Critical Care   31 巻 ( 2 ) 頁: 22 - 26   2019年8月

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    記述言語:日本語   出版者・発行元:一般社団法人 日本神経救急学会  

    <p>救急・ICU管理において脳波モニタリングが有用な疾患として,感染などを契機に発症する急性脳症がある。「二相性発作と遅発性拡散能低下を示す急性脳症(AESD)」と呼ばれる急性脳症は,発症時は熱性けいれん重積と鑑別が困難で,数日後に二相目の発作群発が起こるとともに高度の大脳白質の浮腫が出現する。発症時のMRIでは異常がみられないため,早期の熱性けいれんとの鑑別のために脳波が重要である。またICUで鎮静下に治療を行うため臨床観察のみでは発作の診断が困難で,脳波モニタリングが治療の指標になる。近年は急性脳症に対して低体温療法を試みることが増えており,低体温療法中の脳波所見も知っておく必要がある。これらのICU脳波モニタリングにはamplitude-integrated EEGやdense spectral arrayなどのトレンドグラムが有用である。</p>

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  24. Hippocampal diffusion abnormality after febrile status epilepticus is related to subsequent epilepsy

    Yokoi Setsuri, Kidokoro Hiroyuki, Yamamoto Hiroyuki, Ohno Atsuko, Nakata Tomohiko, Kubota Tetsuo, Tsuji Takeshi, Morishita Masashi, Kawabe Takashi, Naiki Misako, Maruyama Koichi, Itomi Kazuya, Kato Toru, Ito Komei, Natsume Jun

    EPILEPSIA   60 巻 ( 7 ) 頁: 1306 - 1316   2019年7月

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    記述言語:日本語   出版者・発行元:Epilepsia  

    Objective: To assess hippocampal signal changes on diffusion-weighted imaging (DWI) during the acute period after febrile status epilepticus (FSE) and to examine the relationship between DWI and subsequent epilepsy. Methods: A prospective, multicenter study of children with a first episode of FSE was performed. The patients underwent magnetic resonance imaging (MRI) within 3 days of FSE, and signal intensity was evaluated on DWI. Electroencephalography studies within 3 days of FSE were also assessed. Nine to 13 years after FSE, information on subsequent epilepsy was obtained. Results: Twenty-two children with FSE were evaluated. DWI showed unilateral hippocampal hyperintensity in six patients (27%). Three of six patients with hippocampal hyperintensity had ipsilateral thalamic hyperintensity. On EEG within 3 days of FSE, five of six patients with hippocampal hyperintensity had ipsilateral focal slowing, spikes, or attenuation. Nine to 13 years later, the outcomes could be determined in five patients with hippocampal hyperintensity and in 10 without. All 5 patients with hippocampal hyperintensity had hippocampal atrophy and developed focal epilepsy, whereas only 1 of 10 patients without hippocampal hyperintensity developed epilepsy (P = 0.002). Ictal semiology was concordant with temporal lobe seizures in all patients. Ipsilateral temporal epileptiform abnormalities were seen on EEG in four of five at last follow-up. Significance: Acute DWI hippocampal hyperintensity was seen in 27% of patients with FSE. Acute DWI hyperintensity suggests cytotoxic edema caused by prolonged seizure activity. Hippocampal DWI hyperintensity is related to mesial temporal lobe epilepsy and can be a target of neuroprotective treatments to prevent the onset of epilepsy.

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  25. Novel non-invasive modalities for presurgical evaluation in focal epilepsy

    Ito Yuji, Yamamoto Hiroyuki, Okanishi Tohru, Maesawa Satoshi, Natsume Jun

    PEDIATRICS INTERNATIONAL   61 巻 ( 3 ) 頁: 319 - 321   2019年3月

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    出版者・発行元:Pediatrics International  

    DOI: 10.1111/ped.13793

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  26. 小児の急性脳症, 熱性けいれん重積における脳波

    夏目 淳, 大野 敦子, 山本 啓之, 城所 博之, 沼口 敦

    臨床神経生理学   47 巻 ( 1 ) 頁: 53 - 57   2019年2月

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    記述言語:日本語   出版者・発行元:一般社団法人 日本臨床神経生理学会  

    <p>救急・ICU管理において脳波モニタリングが有用な疾患として, 感染などを契機に発症する急性脳症がある。「二相性発作と遅発性拡散能低下を示す急性脳症 (AESD) 」と呼ばれる急性脳症は, 発症時は熱性けいれん重積と鑑別が困難で, 数日後に二相目の発作群発が起こるとともに高度の大脳白質の浮腫が出現する。発症時のMRIでは異常がみられないため, 早期の熱性けいれんとの鑑別のために脳波が重要である。またICUで鎮静下に治療を行うため臨床観察のみでは発作の診断が困難で, 脳波モニタリングが治療の指標になる。近年は急性脳症に対して低体温療法を試みることが増えており, 低体温療法中の脳波所見も知っておく必要がある。これらのICU脳波モニタリングにはamplitude-integrated EEGやdense spectral arrayなどのトレンドグラムが有用である。</p>

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  27. Anatomo-electro-clinical correlations of hypermotor seizures with amygdala enlargement: Hippocampal seizure origin identified using stereoelectroencephalography

    Ishizaki Tomotaka, Maesawa Satoshi, Nakatsubo Daisuke, Yamamoto Hiroyuki, Shibata Masashi, Kato Sachiko, Yoshida Mari, Natsume Jun, Hoshiyama Minoru, Wakabayashi Toshihiko

    EPILEPSY & BEHAVIOR CASE REPORTS   11 巻   頁: 10 - 13   2019年

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    記述言語:日本語   出版者・発行元:Epilepsy and Behavior Case Reports  

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  28. Hereditary cardiac amyloidosis associated with Pro24Ser transthyretin mutation: a case report.

    Yamamoto H, Hashimoto T, Kawamura S, Hiroe M, Yamashita T, Ando Y, Yokochi T

    Journal of medical case reports   12 巻 ( 1 ) 頁: 370   2018年12月

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  29. Longitudinal Findings of MRI and PET in West Syndrome with Subtle Focal Cortical Dysplasia

    Sakaguchi Y., Kidokoro H., Ogawa C., Okai Y., Ito Y., Yamamoto H., Ohno A., Nakata T., Tsuji T., Nakane T., Kawai H., Kato K., Naganawa S., Natsume J.

    AMERICAN JOURNAL OF NEURORADIOLOGY   39 巻 ( 10 ) 頁: 1932 - 1937   2018年10月

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    記述言語:日本語   出版者・発行元:American Journal of Neuroradiology  

    BACKGROUND AND PURPOSE: Despite the development of neuroimaging, identification of focal cortical dysplasia remains challenging. The purpose of this study was to show the longitudinal changes of MR imaging and FDG-PET in patients with West syndrome and subtle focal cortical dysplasia. MATERIALS AND METHODS: Among 52 consecutive patients with West syndrome, 4 were diagnosed with subtle focal cortical dysplasia on 3T MR imaging. MR imaging and PET findings were evaluated longitudinally at onset and at 12 and 24 months of age. RESULTS: At the onset of West syndrome, MR imaging demonstrated focal signal abnormalities of the subcortical white matter in 2 patients. In the other 2 patients, focal subcortical high-intensity signals became visible on follow-up T2WI as myelination progressed. PET at onset showed focal cortical hypometabolism in 3 patients, with 1 of these patients also having focal hypermetabolism and 1 having normal findings. On PET at 24 months, hypometabolism persisted in 2 patients and disappeared in 1, and hypermetabolism disappeared in 1. In 1 patient with normal MR imaging and PET findings at onset, focal hyperintensity and hypometabolism first appeared at 24 months of age. The findings on MR imaging and PET in these patients evolved differently with brain maturation and the clinical course. CONCLUSIONS: Subtle focal cortical dysplasia can be undetectable on MR imaging at the onset of West syndrome and is not always accompanied by hypometabolism or hypermetabolism on PET. Longitudinal MR imaging and PET studies may be useful for detecting such lesions. Even in West syndrome with a congenital structural abnormality, PET findings evolve differently with brain maturation and the clinical condition.

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  30. Long-term follow up of an adult with alternating hemiplegia of childhood and a p.Gly755Ser mutation in the ATP1A3 gene.

    Ito T, Narugami M, Egawa K, Yamamoto H, Asahina N, Kohsaka S, Ishii A, Hirose S, Shiraishi H

    Brain & development   40 巻 ( 3 ) 頁: 226 - 228   2018年3月

  31. Cytotoxic edema at onset in West syndrome of unknown etiology: A longitudinal diffusion tensor imaging study

    Ogawa Chikako, Kidokoro Hiroyuki, Fukasawa Tatsuya, Yamamoto Hiroyuki, Ishihara Naoko, Ito Yuji, Sakaguchi Yoko, Okai Yu, Ohno Atsuko, Nakata Tomohiko, Azuma Yoshiteru, Hattori Ayako, Kubota Tetsuo, Tsuji Takeshi, Hirakawa Akihiro, Kawai Hisashi, Natsume Jun

    EPILEPSIA   59 巻 ( 2 ) 頁: 440 - 448   2018年2月

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    記述言語:日本語   出版者・発行元:Epilepsia  

    Objective: To clarify longitudinal changes in white matter microstructures from the onset of disease in patients with West syndrome (WS) of unknown etiology. Methods: Diffusion tensor imaging (DTI) was prospectively performed at onset and at 12 and 24 months old in 17 children with WS of unknown etiology. DTI was analyzed using tract-based spatial statistics (TBSS) and tract-specific analysis (TSA) of 13 fiber tracts, and fractional anisotropy (FA) and mean diffusivity (MD) were compared with those of 42 age-matched controls. Correlations of FA and MD with developmental quotient (DQ) at age 24 months were analyzed. Multiple comparisons were adjusted for using the false discovery rate (q-value). Results: TBSS analysis at onset showed higher FA and lower MD in the corpus callosum and brainstem in patients. TSA showed lower MD in bilateral uncinate fasciculi (UF) (right: q < 0.001; left: q = 0.03) at onset in patients. TBSS showed a negative correlation between FA at onset and DQ in the right frontal lobe, whereas FA at 24 months old exhibited a positive correlation with DQ in the diffuse white matter. MD for bilateral UF at 24 months old on TSA correlated positively with DQ (q = 0.04, both). Significance: These findings may indicate the existence of cytotoxic edema in the immature white matter and dorsal brainstem at onset, and subsequent alterations in the diffuse white matter in WS of unknown etiology. Microstructural development in the UF might play important roles in cognitive development in WS.

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  32. Paroxysmal nonepileptic events in children with epilepsy

    Ito Yuji, Kidokoro Hiroyuki, Negoro Tamiko, Tanaka Masaharu, Okai Yu, Sakaguchi Yoko, Ogawa Chikako, Takeuchi Tomoya, Ohno Atsuko, Yamamoto Hiroyuki, Nakata Tomohiko, Maesawa Satoshi, Watanabe Kazuyoshi, Takahashi Yoshiyuki, Natsume Jun

    EPILEPSY RESEARCH   132 巻   頁: 59 - 63   2017年5月

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    記述言語:日本語   出版者・発行元:Epilepsy Research  

    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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  33. A study of computer-aided diagnosis for pulmonary nodule: comparison between classification accuracies using calculated image features and imaging findings annotated by radiologists.

    Kawagishi M, Chen B, Furukawa D, Sekiguchi H, Sakai K, Kubo T, Yakami M, Fujimoto K, Sakamoto R, Emoto Y, Aoyama G, Iizuka Y, Nakagomi K, Yamamoto H, Togashi K

    International journal of computer assisted radiology and surgery   12 巻 ( 5 ) 頁: 767 - 776   2017年5月

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  34. White Matter Abnormality Correlates with Developmental and Seizure Outcomes in West Syndrome of Unknown Etiology

    Natsume J., Ogawa C., Fukasawa T., Yamamoto H., Ishihara N., Sakaguchi Y., Ito Y., Takeuchi T., Azuma Y., Ando N., Kubota T., Tsuji T., Kawai H., Naganawa S., Kidokoro H.

    AMERICAN JOURNAL OF NEURORADIOLOGY   37 巻 ( 4 ) 頁: 698 - 705   2016年4月

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    記述言語:日本語   出版者・発行元:American Journal of Neuroradiology  

    BACKGROUND AND PURPOSE: West syndrome is an epileptic encephalopathy characterized by epileptic spasms, a specific pattern on electroencephalography of hypsarrhythmia, and developmental regression. Our aim was to assess white matter abnormalities in West syndrome of unknown etiology. We hypothesized that diffusion tensor imaging reveals white matter abnormalities, especially in patients with poor seizure and developmental outcomes. MATERIALS AND METHODS: We enrolled 23 patients with new-onset West syndrome of unknown etiology. DTI was performed at 12 and 24 months of age. Fractional anisotropy images were compared with those of controls by using tract-based spatial statistics. We compared axial, radial, and mean diffusivity between patients and controls in the fractional anisotropy skeleton. We determined correlations of these parameters with developmental quotient, electroencephalography, and seizure outcomes. We also compared DTI with hypometabolism on fluorodeoxyglucose positron-emission tomography. RESULTS: At 12 months of age, patients showed widespread fractional anisotropy reductions and higher radial diffusivity in the fractional anisotropy skeleton with a significant difference on tract-based spatial statistics. The developmental quotient at 12 months of age correlated positively with fractional anisotropy and negatively with radial and mean diffusivity. Patients with seizure and abnormal findings on electroencephalography after initial treatments had lower fractional anisotropy and higher radial diffusivity. At 24 months, although tract-based spatial statistics did not show significant differences between patients and controls, tract-based spatial statistics in the 10 patients with a developmental quotient of <70 had significant fractional anisotropy reduction. In patients with unilateral temporal lobe hypometabolism on PET, tract-based spatial statistics showed greater fractional anisotropy reduction in the temporal lobe ipsilateral to the side of PET hypometabolism. CONCLUSIONS: Diffuse abnormal findings on DTI at 12 months of age suggest delayed myelination as a key factor underlying abnormal findings on DTI. Conversely, asymmetric abnormal findings on DTI at 24 months may reflect underlying focal pathologies.

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  35. Increased fetal heart rate variability in periventricular leukomalacia

    Kurahashi Hirokazu, Okumura Akihisa, Kubota Tetsuo, Kidokoro Hiroyuki, Maruyama Koichi, Hayakawa Masahiro, Itakura Atsuo, Matsuzawa Katsuji, Yamamoto Hiroyuki, Kato Toru, Hayakawa Fumio, Watanabe Kazuyoshi

    BRAIN & DEVELOPMENT   38 巻 ( 2 ) 頁: 196 - 203   2016年2月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    Objective: This study used quantitative analysis to determine whether increased variability in fetal heart rate (FHR) is related to the risk of developing periventricular leukomalacia (PVL). Methods: We analyzed 124 FHR traces of neonates delivered preterm at 27-33. weeks' gestation to 105 mothers. FHR traces 1-3. h before delivery were translated into power-spectrum curves using a fast Fourier transformation. The total power (the area under the curve of 1-10 cycles per minute), segmental power of every cycle per minute, peak power, and frequency edges were calculated, and their relationship with the subsequent development of PVL was examined. Results: Total power was significantly higher in the PVL group (n= 9, median 1813, range 1064-2426) compared to the non-PVL group (n= 114, median 1383, range 381-3324, p= 0.029). Infants in the PVL group had greater segmental power in segments with 1-2, 2-3, and 9-10 cycles per minute, than those in the non-PVL group. Total power of ≥1550 was significantly correlated with the subsequent development of PVL and premature rupture of membranes. Furthermore, the frequency of pregnancy-induced hypertension was significantly reduced in the fetuses with a total power of ≥1550. Conclusion: Our study suggests that a fetus with increased FHR variability is at risk of developing PVL. This study provides additional evidence supporting the contribution of antenatal factors to the subsequent development of PVL.

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  36. Autopsy findings of a patient with acute encephalitis and refractory, repetitive partial seizures

    Ogawa Chikako, Natsume Jun, Yamamoto Hiroyuki, Ishihara Naoko, Tashiro Atsushi, Kidokoro Hiroyuki, Negoro Tamiko, Yoshida Mari, Watanabe Kazuyoshi

    SEIZURE-EUROPEAN JOURNAL OF EPILEPSY   35 巻   頁: 80 - 82   2016年2月

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    記述言語:日本語   出版者・発行元:Seizure  

    DOI: 10.1016/j.seizure.2016.01.005

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  37. Clinical and neuroimaging findings in children with posterior reversible encephalopathy syndrome

    Yamamoto Hiroyuki, Natsume Jun, Kidokoro Hiroyuki, Ishihara Naoko, Suzuki Motomasa, Tsuji Takeshi, Kubota Tetsuo, Yamada Akio, Ozeki Michio, Kato Zenichiro, Kawamura Yoshiki, Yoshikawa Tetsushi, Okumura Akihisa, Ando Naoki, Saitoh Shinji, Takahashi Yoshiyuki, Watanabe Kazuyoshi, Kojima Seiji

    EUROPEAN JOURNAL OF PAEDIATRIC NEUROLOGY   19 巻 ( 6 ) 頁: 672 - 678   2015年11月

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    記述言語:日本語   出版者・発行元:European Journal of Paediatric Neurology  

    Objective To clarify the clinical and radiological spectrum of posterior reversible encephalopathy syndrome (PRES) in children, and to identify the prognostic factors. Methods The records of 40 children with PRES were reviewed. Acute clinical symptoms, MRI including apparent diffusion coefficient (ADC) maps in the acute and follow-up periods and neurological sequelae, including epilepsy, were noted. Results Age at onset ranged from 2 to 16 years. Underlying disorders were hematological or neoplastic disorders (n = 20), renal diseases (n = 14) and others (n = 6). In the acute period, 31 patients had seizures, 25 had altered consciousness, 11 had visual disturbances and 10 had headache. Of 29 patients who had ADC maps in the acute period, 13 had reduced diffusivity as shown by ADC within PRES lesions. Of 26 patients with follow-up MRI, 13 had focal gliosis or cortical atrophy. No patients had motor impairment, and four patients had focal epilepsy. No clinical variables were associated with focal gliosis or cortical atrophy on follow-up MRI, but lesional ADC reduction in the acute period was prognostic for focal gliosis or cortical atrophy on follow-up MRI (p = 0.005). Conclusions To the best of our knowledge, this is the largest cohort study to date involving PRES in children. Acute symptoms in pediatric patients are similar to those reported in adults, but altered consciousness was more frequent in children. Lesional ADC reduction in the acute period was common and was a good predictor of later, irreversible MRI lesions.

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  38. TUBA1A mutation can cause a hydranencephaly-like severe form of cortical dysgenesis

    Yokoi Setsuri, Ishihara Naoko, Miya Fuyuki, Tsutsumi Makiko, Yanagihara Itaru, Fujita Naoko, Yamamoto Hiroyuki, Kato Mitsuhiro, Okamoto Nobuhiko, Tsunoda Tatsuhiko, Yamasaki Mami, Kanemura Yonehiro, Kosaki Kenjiro, Kojima Seiji, Saitoh Shinji, Kurahashi Hiroki, Natsume Jun

    SCIENTIFIC REPORTS   5 巻   頁: 15165   2015年10月

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    記述言語:日本語   出版者・発行元:Scientific Reports  

    TUBA1A mutations cause a wide spectrum of lissencephaly and brain malformations. Here, we report two patients with severe cortical dysgeneses, one with an extremely thin cerebral parenchyma apparently looking like hydranencephaly and the other with lissencephaly accompanied by marked hydrocephalus, both harbouring novel de novo missense mutations of TUBA1A. To elucidate how the various TUBA1A mutations affect the severity of the phenotype, we examined the capacity of the mutant protein to incorporate into the endogenous microtubule network in transfected COS7 cells by measuring line density using line extraction in an immunofluorescence study. The mutants responsible for severe phenotypes were found to incorporate extensively into the network. To determine how each mutant alters the microtubule stability, we examined cold-induced microtubule depolymerisation in fibroblasts. The depolymerisation of patients' fibroblasts occurred earlier than that of control fibroblasts, suggesting that microtubules bearing mutated tubulins are unstable. Both mutations are predicted to participate in lateral interactions of microtubules. Our data suggest that the TUBA1A mutations disrupting lateral interactions have pronounced dominant-negative effects on microtubule dynamics that are associated with the severe end of the lissencephaly spectrum.

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  39. Diffusion-weighted MRI for early diagnosis of neonatal herpes simplex encephalitis

    Okanishi Tohru, Yamamoto Hiroyuki, Hosokawa Takatoshi, Ando Naoki, Nagayama Yoshihisa, Hashimoto Yuji, Maihara Toshiro, Goto Tomohide, Kubota Tetsuo, Kawaguchi Chiharu, Yoshida Hiroshi, Sugiura Katsumi, Itomi Seiko, Ohno Koyo, Takanashi Jun-ichi, Hayakawa Masahiro, Otsubo Hiroshi, Okumura Akihisa

    BRAIN & DEVELOPMENT   37 巻 ( 4 ) 頁: 423 - 431   2015年4月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    Aim: To determine the early changes and evolutions of brain diffusion-weighted imaging (DWI), and analyze prognostic factors of the early changes among patients with neonatal herpes simplex encephalitis (NHSE). Method: We selected patients who developed encephalitis by 28. d after birth; had herpes simplex infection; and who underwent magnetic resonance imaging, including DWI, ≤7. d of symptom onset. Thirty-two DWI scans between 0 and 28. d after onset in 13 patients and the clinical data were recruited. The distribution, evolution of the lesions, and neurological outcome were analyzed. Results: DWI frequently showed multiple cortical lesions in both hemispheres in the early period and both hemispheres on DWI (8/9 scans at ≤48. h, 7/7 patients). As time from onset increased, the cortical lesions tended to coincide with subcortical white matter lesions beneath the initial cortical lesions (p<. 0.01). Lesions from the cortex extended to the subcortical white matter in 7 patients. Deep cerebral lesions, involving basal ganglia, internal capsules, thalamus, were also found in 9 patients ≤7. d of onset. The distributions of deep cerebral lesions (none/unilateral/bilateral) ≤7. d of onset showed significant correlations with neurological prognoses (gross motor functions: p<. 0.01; developmental or intellectual quotient scores: p<. 0.01). Interpretation: Cortical lesions were main findings of DWI in NHSE in the early period. Bilateral deep cerebral lesions ≤7. d were highly indicative of poor motor and cognitive outcomes.

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  40. A severity score for acute necrotizing encephalopathy

    Yamamoto Hiroyuki, Okumura Akihisa, Natsume Jun, Kojima Seiji, Mizuguchi Masashi

    BRAIN & DEVELOPMENT   37 巻 ( 3 ) 頁: 322 - 327   2015年3月

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    記述言語:日本語   出版者・発行元:Brain and Development  

    Objective: To develop a score that predicts the prognosis of children with acute necrotizing encephalopathy (ANE). Method: We retrospectively evaluated clinical variables and neurological outcome in two cohorts of children with ANE. Firstly, we developed the ANE severity score (ANE-SS) according to the clinical variables that correlated with neurological outcome in 41 children who were included in our previous reports in 2009. We then applied the scoring system to a second cohort of 32 patients who were newly collected in 2011. We investigated the correlation between the ANE-SS and neurological outcome in all 73 patients. Results: In the first cohort, brain stem lesions on MRI and state of shock at onset were significantly correlated with outcome. Age over 48. months, elevated CSF protein, and low platelet counts tended to be correlated with outcome. No types of treatment were correlated with outcome. The developed ANE-SS ranged from 0 to 9 points, with 3 points for existence of shock, 2 points for brain stem lesions, 2 points for age over 48. months, 1 point for platelet count below 100,000/μL, and 1 point for CSF protein above 60. mg/dl. Patients were classed as low risk (ANE-SS 0-1 points), medium risk (ANE-SS 2-4 points), or high risk (ANE-SS 5-9 points). ANE-SS was significantly correlated with outcome in the group of 73 patients. Conclusion: ANE-SS can be used to predict outcome in patients with ANE. More effective treatments need to be developed for high-risk patients.

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講演・口頭発表等 6

  1. COVID-19流行下における急性脳炎脳症発症数変化

    山本啓之

    第64回日本小児神経学会学術集会  2022年6月5日  日本小児神経学会

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    開催年月日: 2022年6月

    記述言語:日本語   会議種別:口頭発表(一般)  

    開催地:高崎   国名:日本国  

  2. 初学者のための 新生児脳波の実際 招待有り

    山本啓之

    第51回日本臨床神経生理学会学術大会  2021年12月17日  日本臨床神経生理学会

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    開催年月日: 2021年12月

    記述言語:日本語   会議種別:公開講演,セミナー,チュートリアル,講習,講義等  

    開催地:仙台   国名:日本国  

  3. 当院の best of the best   時間分解能を活かしたLandau-Kleffner症候群の脳磁図解析 招待有り

    山本啓之

    第51回日本臨床神経生理学会学術大会  2021年12月18日  日本臨床神経生理学会

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    開催年月日: 2021年12月

    記述言語:日本語   会議種別:シンポジウム・ワークショップ パネル(指名)  

    開催地:仙台   国名:日本国  

  4. Landau-Kleffner症候群 1例の脳磁図解析

    山本啓之

    第54回日本てんかん学会学術集会  2021年9月24日  日本てんかん学会

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    開催年月日: 2021年9月

    記述言語:日本語   会議種別:ポスター発表  

    開催地:名古屋   国名:日本国  

  5. 愛知県てんかん診療コンソーシアム におけるコーディネーターの役割 招待有り

    山本啓之

    第54回日本てんかん学会学術集会  2021年9月23日  日本てんかん学会

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    開催年月日: 2021年9月

    記述言語:日本語   会議種別:シンポジウム・ワークショップ パネル(指名)  

    開催地:名古屋   国名:日本国  

  6. 2018-2019年度における 急性脳炎脳症発症数の比較

    山本啓之

    第63回日本小児神経学会学術集会  2021年5月28日  日本小児神経学会

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    開催年月日: 2021年5月

    記述言語:日本語   会議種別:ポスター発表  

    開催地:福岡   国名:日本国  

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科研費 2

  1. 脳磁場計測による小児急性脳炎脳症後の脳神経ネットワークの解明

    研究課題/研究課題番号:20K16851  2020年4月 - 2024年3月

    科学研究費助成事業  若手研究

    山本 啓之

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    担当区分:研究代表者 

    配分額:3900000円 ( 直接経費:3000000円 、 間接経費:900000円 )

    小児急性脳炎脳症は生命予後は改善してきたが、機能予後の改善が十分に得られているとは言えず、高次脳機能障害を残す症例も多い。高次機能障害には発達過程でわかってくる障害が含まれる。しかし、脳症後の高次脳機能障害を客観的・定量的に評価する手法は確立していない。脳磁図は高い信号源推定能精度と高い時間分解能を有し、脳神経活動を直接測定する機器である。本研究では、認知機能に関する脳神経ネットワークにおける正常小児の発達に伴う変化および急性脳炎脳症患児の機能障害の検出および障害部位を明らかとし、脳症後の高次脳機能障害の早期検出と治療効果の判定に役立てることを目的とする。
    小児急性脳炎脳症は2000年代以降、生命予後は改善してきた。しかし、機能予後の改善が十分に得られているとは言えず、後遺症として高次脳機能障害を残す症例が多い。後遺症としての高次機能障害には、重症例に加え、その後の発達過程で顕在化する潜在性の障害が含まれる。しかし、脳症後の高次脳機能障害を客観的・定量的に評価する手法は確立していない。また、高次機能障害の定量的評価を行うことができれば、潜在的な脳機能障害を検出し、機能障害への早期の治療的介入や、現行および新規に開発される治療の効果を判定することが可能となる。脳磁図は高い信号源推定能精度と高い時間分解能を有し、脳神経活動を直接測定する機器である。近年の解析技術の進歩により、脳神経活動のネットワーク解析や認知情報処理パターンの解析が可能となってきている。しかし、脳神経活動のネットワークの解析技術を小児領域の認知機能障害に用いた報告はほとんど見られず、本領域での研究が待たれている現状である。本研究では、認知機能に関する脳神経ネットワークにおける正常小児の発達に伴う変化および急性脳炎脳症患児の機能障害の検出および障害部位を明らかとし、脳症後の高次脳機能障害の早期検出と治療効果の判定に役立てることを目的とする。
    これにより成人の加齢による報告と同様に小児は発達により、脳神経ネットワークが変化していくことが予想される。その年齢ごとに急性脳炎脳症患児のネットワーク障害部位が明らかとなることでリハビリや教育などの介入の参考となる。また、現在の治療では根拠をもった治療法は開発されておらず治療による障害の違いはないことが予想される。将来的に、有効と考えられる治療が開発された際には本研究の手法を用いて治療効果判定を行うことが可能となると考える。
    本研究は被験者の協力が必須であるが、新型コロナウイルスの流行により被験者を受け入れられない期間が続いたため、実質的に磁場計測ができていない。正確な磁場計測のための準備や環境整備に終始している。
    感染対策を徹底したうえで、新型コロナウイルス感染症の流行下においても脳磁計設置機関との調整を行いすすめていく予定である。

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  2. 脳磁図によるウェスト症候群のてんかん焦点の解明と予後予測

    研究課題/研究課題番号:17K16250  2017年4月 - 2020年3月

    科学研究費助成事業  若手研究(B)

    山本 啓之

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    担当区分:研究代表者 

    配分額:2210000円 ( 直接経費:1700000円 、 間接経費:510000円 )

    ウェスト症候群14名に計20回の脳磁計測を行った。全例でhypsarrythmiaというウェスト症候群に特徴的な脳波所見を呈し、全例で間欠的なhypsarrhthmiaであった。間欠的hypsarrhythmiaの起始部のみの解析を行った。脳腫瘍に伴う症候性ウェスト症候群の1例において、dSPM解析で腫瘍周囲から高い磁場変化が発生し、前頭葉へと拡延することが確認された。他のウェスト症候群においてはsingle dipole法、dSPM法双方で有意な所見は得られなかった。前例で発作予後、発達予後に関して追跡調査中であり、今後新たな知見が得られる可能性がある。
    ウェスト症候群はhypsarrhythmiaという特徴的な脳波所見を呈し、その所見を早期に改善させることが発達予後の改善につながることがわかっている。しかしhypsarrhmiaが脳のどこから発生し、何を意味しているかはほとんど解明されていない。今回脳波よりも焦点推定能の高い脳磁計を用いて磁場計測できたことでその解明につながる基礎的な情報を得ることができた。また、一礼では焦点推定が可能であったため、脳外科手術につなげることができ、発作、発達予後の改善につながることが判明した。今後も一部のウェスト症候群では焦点推定により発作、発達予後の改善できる症例があることが予想された。

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社会貢献活動 1

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