Updated on 2022/05/20

写真a

 
YAMAMOTO Hiroyuki
 
Organization
Nagoya University Hospital Pediatrics Assistant Professor
Graduate School
Graduate School of Medicine
Title
Assistant Professor
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Degree 1

  1. 博士(医学) ( 2016.1   名古屋大学 ) 

Research Areas 1

  1. Life Science / Embryonic medicine and pediatrics  / child neurology, epilepsy, neurophysiology

Research History 9

  1. Nagoya University   Assistant Professor

    2019.9

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  2. Nagoya University

    2016.4 - 2019.8

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  3. Hokkaido University   Researcher

    2015.4 - 2016.3

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  4. 愛知県青い鳥医療福祉センター   小児科

    2014.4 - 2015.3

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  5. Nagoya University

    2010.10 - 2014.3

  6. 愛知県厚生連安城更生病院   小児科

    2009.9 - 2010.9

  7. Nagoya University

    2009.4 - 2009.9

  8. 愛知県心身障害者コロニー中央病院   新生児科

    2008.4 - 2009.3

  9. 名古屋掖済会病院   初期研修医

    2004.4 - 2006.3

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Education 2

  1. 名古屋大学大学院医学系研究科   健康社会医学専攻

    2010.4 - 2014.3

  2. Nagoya University   School of Medicine

    1998.4 - 2004.3

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    Country: Japan

Professional Memberships 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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Committee Memberships 2

  1. 日本てんかん学会   てんかん学教育委員  

    2021.10   

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    Committee type:Academic society

  2. 日本てんかん学会   法的問題検討委員会  

    2021.10   

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    Committee type:Academic society

 

Papers 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   Vol. 44 ( 3 ) page: 221 - 228   2022.3

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    Language:Japanese   Publisher: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   Vol. 64 ( 1 ) page: e15001   2022.1

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    Language:Japanese   Publisher: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   Vol. 125   page: 108397   2021.12

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    Language:Japanese   Publisher: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   Vol. 43 ( 10 ) page: 979 - 987   2021.11

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    Language:Japanese   Publisher: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.

    DOI: 10.1016/j.braindev.2021.07.003

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

    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   Vol. 121 ( Pt A ) page: 108075   2021.8

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    Language:Japanese   Publisher: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.

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

    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   Vol. 79   page: 38 - 44   2021.6

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    Language:Japanese   Publisher: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

    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   Vol. 118   page: 107924   2021.5

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    Language:Japanese   Publisher: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   Vol. 63 ( 5 ) page: 761 - 768   2021.5

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    Language:Japanese   Publisher: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 International journal

    BRAIN & DEVELOPMENT   Vol. 43 ( 2 ) page: 181 - 185   2021.2

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    Language:Japanese   Publisher:Brain and Development  

    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   Vol. 43 ( 1 ) page: 69 - 77   2021.1

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    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. A case report of neonatal seizures in which fosphenytoin was effective

    Hashimoto Misa, Kanzawa Takahiro, Yamamoto Hiroyuki, Kato Eiko, Ieda Kuniko

    Journal of Japan Society of Perinatal and Neonatal Medicine   Vol. 57 ( 1 ) page: 124 - 128   2021

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    Language:Japanese   Publisher:Japan Society of Perinatal and Neonatal Medicine  

    <p> In Japan, phenobarbital(PB)is recommended as the first-line agent and midazolam(MDL)is recommended as the second-line agent for treatment for neonatal seizures. However, there is not sufficient evidence to determine the optimal agent for neonatal seizures for which PB and MDL are not effective. We report a case in which fosphenytoin(fos-PHT)was effective for neonatal seizures. The male infant was born at 40 weeks by vacuum extraction due to a non-reassuring fetal status. His Apgar score was 1 at 1 minute, 4 at 5 minutes, and 5 at 10 minutes, and he was admitted to Neonatal Intensive Care Unit with suspected neonatal asphyxia. On 32 hours after his birth, the amplitude-integrated EEG waveform showed a saw-tooth pattern, and the same type of waveform was observed for approximately 30 seconds on the simultaneously recorded EEG. Following this EEG abnormality, he had clonic seizures in his left upper limb and we had diagnosed neonatal seizures. The seizures persisted after administration of PB and MDL, and only disappeared after the administration of fos-PHT. No side effects such as hypotension or bradycardia due to the fos-PHT were observed. His parents were informed of this off label use of fos-PHT for their infant and agreed to our proposal. We recognized that fos-PHT was effective and safe drug for neonatal seizures.</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

    PEDIATRIC NEUROLOGY   Vol. 111   page: 78 - 84   2020.10

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    Language:Japanese   Publisher:Pediatric Neurology  

    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   Vol. 131 ( 9 ) page: 2100 - 2104   2020.9

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    Language:Japanese   Publisher: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.

    DOI: 10.1016/j.clinph.2020.06.006

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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   Vol. 27   page: 30 - 36   2020.7

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    Language:Japanese   Publisher: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.

    DOI: 10.1016/j.ejpn.2020.05.004

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

    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   Vol. 134 ( 4 ) page: 1027 - 1036   2020.4

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    Language:Japanese   Publisher: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.

    DOI: 10.3171/2020.1.JNS192567

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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   Vol. 10 ( 1 ) page: 5263   2020.3

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    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   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.

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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   Vol. 42 ( 2 ) page: 217 - 221   2020.2

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    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   Vol. 103 ( Pt A ) page: 106359   2020.2

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    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. Introduction

    Yamamoto Hiroyuki, Maegaki Yoshihiro

    NO TO HATTATSU   Vol. 52 ( 4 ) page: 238 - 239   2020

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    DOI: 10.11251/ojjscn.52.238

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  21. Treatment of acute encephalopathy with EEG monitoring

    Yamamoto Hiroyuki

    NO TO HATTATSU   Vol. 52 ( 4 ) page: 246 - 248   2020

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    DOI: 10.11251/ojjscn.52.246

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

    Kumai Sumire, Yamamoto Hiroyuki, Nakata Tomohiko, Kidokoro Hiroyuki, Fujiura Naoko, Shibata Motohiro, Kaneko Kimihiko, Takahashi Toshiyuki, Natsume Jun

    NO TO HATTATSU   Vol. 52 ( 6 ) page: 414 - 418   2020

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    <p>  Myelin oligodendrocyte glycoprotein antibody (MOG-Ab) is identified in some patients with ADEM. Whether serial change of MOG-Ab 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-year-old 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.</p>

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  23. Continuous EEG monitoring in children with acute encephalopathy

    NATSUME JUN, OHNO ATSUKO, YAMAMOTO HIROYUKI, KIDOKORO HIROYUKI, NUMAGUCHI ATSUSHI

    Journal of Japan Society of Neurological Emergencies & Critical Care   Vol. 31 ( 2 ) page: 22 - 26   2019.8

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    <p>It is important to detect non-convulsive seizures in the treatment of critically ill children in ICU. Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is characterized by a febrile status epilepticus as the initial neurological symptom, followed by secondary seizures and subcortical white matter edema at day 4 to 6. As MRI shows no acute abnormality at the onset, EEG is useful to distinguish AESD from febrile status epilepticus in early stage. Recently therapeutic hypothermia is applied as a treatment of AESD, and continuous EEG monitoring during the therapy is also important. Amplitudeintegrated EEG (aEEG) and dense spectral array (DSA) are useful for the identification of non-convulsive seizures in long-term EEG monitoring in ICU.</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   Vol. 60 ( 7 ) page: 1306 - 1316   2019.7

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    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   Vol. 61 ( 3 ) page: 319 - 321   2019.3

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    DOI: 10.1111/ped.13793

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  26. Continuous EEG monitoring in children with acute encephalopathy

    Natsume Jun, Ohno Atsuko, Yamamoto Hiroyuki, Kidokoro Hiroyuki, Numaguchi Atsushi

    Japanese Journal of Clinical Neurophysiology   Vol. 47 ( 1 ) page: 53 - 57   2019.2

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    <p>It is important to detect non-convulsive seizures in the treatment of critically ill children in ICU. Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is characterized by a febrile status epilepticus as the initial neurological symptom, followed by secondary seizures and subcortical white matter edema at day 4 to 6. As MRI shows no acute abnormality at the onset, EEG is useful to distinguish AESD from febrile status epilepticus in early stage. Recently therapeutic hypothermia is applied as a treatment of AESD, and continuous EEG monitoring during the therapy is also important. Amplitude-integrated EEG (aEEG) and dense spectral array (DSA) are useful for the identification of non-convulsive seizures in long-term EEG monitoring in ICU.</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   Vol. 11   page: 10 - 13   2019

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    DOI: 10.1016/j.ebcr.2018.09.011

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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   Vol. 12 ( 1 ) page: 370   2018.12

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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   Vol. 39 ( 10 ) page: 1932 - 1937   2018.10

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    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   Vol. 40 ( 3 ) page: 226 - 228   2018.3

  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   Vol. 59 ( 2 ) page: 440 - 448   2018.2

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    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   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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  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   Vol. 12 ( 5 ) page: 767 - 776   2017.5

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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   Vol. 37 ( 4 ) page: 698 - 705   2016.4

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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   Vol. 38 ( 2 ) page: 196 - 203   2016.2

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    DOI: 10.1016/j.braindev.2015.08.008

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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   Vol. 35   page: 80 - 82   2016.2

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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   Vol. 19 ( 6 ) page: 672 - 678   2015.11

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    DOI: 10.1016/j.ejpn.2015.07.005

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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   Vol. 5   page: 15165   2015.10

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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   Vol. 37 ( 4 ) page: 423 - 431   2015.4

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    DOI: 10.1016/j.braindev.2014.07.006

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  40. A severity score for acute necrotizing encephalopathy

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

    BRAIN & DEVELOPMENT   Vol. 37 ( 3 ) page: 322 - 327   2015.3

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KAKENHI (Grants-in-Aid for Scientific Research) 2

  1. The network analysis of brain after acute encephalopathy using MEG

    Grant number:20K16851  2020.4 - 2024.3

    Grants-in-Aid for Scientific Research  Grant-in-Aid for Early-Career Scientists

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    Grant amount:\3900000 ( Direct Cost: \3000000 、 Indirect Cost:\900000 )

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  2. The focus and prognosis analysis of West syndrome using MEG

    Grant number:17K16250  2017.4 - 2020.3

    Grants-in-Aid for Scientific Research  Grant-in-Aid for Young Scientists (B)

    Yamamoto Hiroyuki

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    Authorship:Principal investigator 

    Grant amount:\2210000 ( Direct Cost: \1700000 、 Indirect Cost:\510000 )

    We analyzed MEG of 14 West syndrome patients, and 20times. All patients showed hypsarrhythmia with periodicity. We analyzed the onset of periodic hypsarrhythmia. One case with brain tumor showed significant magnetic field change around tumor by single dipole method. The dSPM analysis showed the significant magnetic field change occured around tumor and propagated to the frontal lobe. The other thirteen patients showed no specific findings in both of single dipole method analysis and dSPM analysis. All patients are followed regarding prognosis of epilepsy and development by childe neurologists continuously. We continue to investigate the new findings.

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Social Contribution 1

  1. 愛知県てんかん治療医療連携協議会

    Role(s):Organizing member

    愛知県てんかん治療医療連携協議会 

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Media Coverage 1

  1. CBCラジオ「きく!ラジオ」てんかん特集 TV or radio program

    CBCラジオ  「きく!ラジオ」  2021.2

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