研究者詳細 - 柴田　敬

論文 - 柴田　敬

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A retrospective study on post-operative recovery of daily living activity after total corpus callosotomy. 国際誌

Hiroki Tsuchiya, Takashi Shibata, Tatsuya Sasaki, Mari Akiyama, Tomoyuki Akiyama, Katsuhiro Kobayashi

Brain & development 2024年9月

記述言語：英語掲載種別：研究論文（学術雑誌）

INTRODUCTION: Post-operative complications of corpus callosotomy (CC) in children, prolonged hospitalization due to inactivity as acute disconnection syndrome is occasionally experienced. We aimed to clarify this issue and its risk factors with a hypothesis that electroencephalogram (EEG) findings as measures of functional lateralization may be among prognostic factors for post-operative recovery. MATERIALS AND METHODS: Twenty-three patients with childhood-onset drug-resistant epilepsy who underwent total CC between April 2017 and December 2023 were included in the study and they were divided into two groups based on the duration of post-operative hospitalization as an indicator of recovery of daily living activity. We compared scalp EEG findings and the other factors including clinical characteristics between the two groups. RESULTS: Of 22 patients (14 males) without specific complications, post-operative hospitalization clustered in 9-14 days in 15 patients (Group A) with range 16-118 days in seven patients (Group B). The ratios of patients with non-lateralized spikes on pre-operative EEG and that of patients with symmetric background activity on post-operative EEG were significantly greater in Group B (7/7, 100 %; 6/7, 86 %, respectively) than in Group A (8/15, 53 %; 4/15, 27 %, respectively) (p = 0.038; p = 0.020, respectively, by Fisher's exact test), while other factors were not significantly different between the two groups. CONCLUSIONS: Delayed recovery of living activity should be anticipated, especially in patients with non-lateralized epileptic discharges on pre-operative EEG.

DOI： 10.1016/j.braindev.2024.09.006

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8歳MRI陰性側頭葉てんかんの一手術例

佐々木達也, 井本良二, 松田奈央子, 柴田敬, 秋山麻里, 亀田雅博, 安原隆雄, 秋山倫之, 小林勝弘, 伊達勲

てんかん研究 42 ( 1 ) 58 - 59 2024年6月

新生児低血糖症による両側後頭葉病変を背景としたWest症候群の1手術例

竹中暁, 土屋弘樹, 柴田敬, 所谷知穂, 佐々木達也, 伊達勲, 秋山倫之, 小林勝弘

てんかん研究 42 ( 1 ) 64 - 64 2024年6月

Modulation index predicts the effect of ethosuximide on developmental and epileptic encephalopathy with spike-and-wave activation in sleep. 国際誌

Takashi Shibata, Hiroki Tsuchiya, Mari Akiyama, Tomoyuki Akiyama, Katsuhiro Kobayashi

Epilepsy research 202 107359 - 107359 2024年5月

担当区分：筆頭著者記述言語：英語掲載種別：研究論文（学術雑誌）

PURPOSE: In developmental and epileptic encephalopathy with spike-and-wave activation in sleep (DEE-SWAS), the thalamocortical network is suggested to play an important role in the pathophysiology of the progression from focal epilepsy to DEE-SWAS. Ethosuximide (ESM) exerts effects by blocking T-type calcium channels in thalamic neurons. With the thalamocortical network in mind, we studied the prediction of ESM effectiveness in DEE-SWAS treatment using phase-amplitude coupling (PAC) analysis. METHODS: We retrospectively enrolled children with DEE-SWAS who had an electroencephalogram (EEG) recorded between January 2009 and September 2022 and were prescribed ESM at Okayama University Hospital. Only patients whose EEG showed continuous spike-and-wave during sleep were included. We extracted 5-min non-rapid eye movement sleep stage N2 segments from EEG recorded before starting ESM. We calculated the modulation index (MI) as the measure of PAC in pair combination comprising one of two fast oscillation types (gamma, 40-80 Hz; ripples, 80-150 Hz) and one of five slow-wave bands (delta, 0.5-1, 1-2, 2-3, and 3-4 Hz; theta, 4-8 Hz), and compared it between ESM responders and non-responders. RESULTS: We identified 20 children with a diagnosis of DEE-SWAS who took ESM. Fifteen were ESM responders. Regarding gamma oscillations, significant differences were seen only in MI with 0.5-1 Hz slow waves in the frontal pole and occipital regions. Regarding ripples, ESM responders had significantly higher MI in coupling with all slow waves in the frontal pole region, 0.5-1, 3-4, and 4-8 Hz slow waves in the frontal region, 3-4 Hz slow waves in the parietal region, 0.5-1, 2-3, 3-4, and 4-8 Hz slow waves in the occipital region, and 3-4 Hz slow waves in the anterior-temporal region. SIGNIFICANCE: High MI in a wider area of the brain may represent the epileptic network mediated by the thalamus in DEE-SWAS and may be a predictor of ESM effectiveness.

DOI： 10.1016/j.eplepsyres.2024.107359

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Exploration of urine metabolic biomarkers for new-onset, untreated pediatric epilepsy: A gas and liquid chromatography mass spectrometry-based metabolomics study. 査読国際誌

Tomoyuki Akiyama, Daisuke Saigusa, Takushi Inoue, Chiho Tokorodani, Mari Akiyama, Rie Michiue, Atsushi Mori, Eiji Hishinuma, Naomi Matsukawa, Takashi Shibata, Hiroki Tsuchiya, Katsuhiro Kobayashi

Brain & development 2024年1月

記述言語：英語掲載種別：研究論文（学術雑誌）

OBJECTIVE: The discovery of objective indicators for recent epileptic seizures will help confirm the diagnosis of epilepsy and evaluate therapeutic effects. Past studies had shortcomings such as the inclusion of patients under treatment and those with various etiologies that could confound the analysis results significantly. We aimed to minimize such confounding effects and to explore the small molecule biomarkers associated with the recent occurrence of epileptic seizures using urine metabolomics. METHODS: This is a multicenter prospective study. Subjects included pediatric patients aged 2 to 12 years old with new-onset, untreated epilepsy, who had had the last seizure within 1 month before urine collection. Controls included healthy children aged 2 to 12 years old. Those with underlying or chronic diseases, acute illnesses, or recent administration of medications or supplements were excluded. Targeted metabolome analysis of spot urine samples was conducted using gas chromatography (GC)- and liquid chromatography (LC)-tandem mass spectrometry (MS/MS). RESULTS: We enrolled 17 patients and 21 controls. Among 172 metabolites measured by GC/MS/MS and 41 metabolites measured by LC/MS/MS, only taurine was consistently reduced in the epilepsy group. This finding was subsequently confirmed by the absolute quantification of amino acids. No other metabolites were consistently altered between the two groups. CONCLUSIONS: Urine metabolome analysis, which covers a larger number of metabolites than conventional biochemistry analyses, found no consistently altered small molecule metabolites except for reduced taurine in epilepsy patients compared to healthy controls. Further studies with larger samples, subjects with different ages, expanded target metabolites, and the investigation of plasma samples are required.

DOI： 10.1016/j.braindev.2023.12.004

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尿中メタボローム解析を用いた未治療てんかんのバイオマーカー探索

秋山倫之, 三枝大輔, 秋山麻里, 柴田敬, 土屋弘樹, 道上理絵, 小林勝弘

脳と発達 56 ( 1 ) 71 - 71 2024年1月

Effective Epilepsy Surgery for Post-Traumatic West Syndrome Following Abusive Head Trauma.

Hiroki Tsuchiya, Takashi Shibata, Tatsuya Sasaki, Takushi Inoue, Isao Date, Tomoyuki Akiyama, Katsuhiro Kobayashi

Acta medica Okayama 77 ( 5 ) 561 - 566 2023年10月

記述言語：英語

West syndrome, an infantile developmental and epileptic encephalopathy with a deleterious impact on long-term development, requires early treatment to minimize developmental abnormality; in such cases, epilepsy surgery should be considered a powerful therapeutic option. We describe a 10-month-old female admitted with West syndrome associated with a hemispheric lesion following abusive head trauma. Her seizures were suppressed by hemispherotomy at 12 months of age, leading to developmental improvement. Surgical treatment of West syndrome following traumatic brain injury has not been reported previously but is worth considering as a treatment option, depending on patient age and brain plasticity.

DOI： 10.18926/AMO/65980

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West症候群に対するVGBの6年間の使用経験

道上理絵, 土屋弘樹, 柴田敬, 秋山麻里, 秋山倫之, 小林勝弘

てんかん研究 41 ( 2 ) 403 - 403 2023年9月

A female patient with adolescent-onset progressive myoclonus epilepsy carrying a truncating MECP2 mutation. 国際誌

Mari Akiyama, Tomoyuki Akiyama, Hirotomo Saitsu, Yukie Tokioka, Rie Tsukahara, Hiroki Tsuchiya, Takashi Shibata, Katsuhiro Kobayashi

Brain & development 45 ( 10 ) 597 - 602 2023年8月

記述言語：英語

BACKGROUND: MECP2 is a well-known causative gene for Rett syndrome but other phenotypes have also been reported. Here, we report a case of a female patient with adolescent-onset progressive myoclonus epilepsy (PME) carrying a novel truncating mutation in the MECP2 gene. CASE REPORT: The patient was a 29-year-old woman with infantile-onset intellectual disability of unspecified cause. She had demonstrated slow but steady development with moderate intellectual disability until the age of 16, when she started having epileptic seizures. Her epilepsy progressed intractably with multiple seizure types accompanied by myoclonus, tremor, and gradual regression. She is currently apathetic and requires extensive assistance in all aspects of life. After an extensive work-up for underlying diseases for PME turned out negative, whole-exome sequencing revealed a de novo 113-bp deletion and 3-bp insertion in MECP2, a variant of NM_004992.4:c.1099_1211delinsGGG, p.(His367Glyfs*32). CONCLUSIONS: The clinical presentation of this case was inconsistent with Rett syndrome, and the rapid regression in the patient's twenties was considered characteristic. Mutations of MECP2 may result in variable neurodevelopmental phenotypes and may also be considered a causative gene for adolescent-onset PME.

DOI： 10.1016/j.braindev.2023.07.006

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Epilepsy in Children With Congenital Heart Disease: Risk Factors and Characteristic Presentations. 国際誌

Takashi Shibata, Maiko Kondo, Yosuke Fukushima, Mari Akiyama, Tomoyuki Akiyama, Teruko Morooka, Kenji Baba, Shinichi Ohtsuki, Hirokazu Tsukahara, Shingo Kasahara, Katsuhiro Kobayashi

Pediatric neurology 147 28 - 35 2023年7月

担当区分：筆頭著者記述言語：英語掲載種別：研究論文（学術雑誌）

BACKGROUND: Children with a congenital heart disease (CHD) are at a higher risk of developing epilepsy than the general population, but detailed characteristics of CHD-associated epilepsy have not been clarified. The purposes of this study were to determine the risk factors for developing epilepsy associated with CHD and to elucidate the characteristics of such epilepsy. METHODS: We performed a retrospective cohort study based on medical records of pediatric patients with CHD who were born between January 2006 and December 2016, underwent cardiac surgery at Okayama University Hospital, and were followed up until at least age three years. Multivariate logistic regression analysis was used to determine factors particularly associated with epilepsy occurrence. In patients who developed epilepsy, clinical data on seizure characteristics were further investigated. RESULTS: We collected data from 1024 patients, and 41 (4.0%) developed epilepsy. The presence of underlying disease (odds ratio [OR]: 2.413; 95% confidence interval [CI]: 1.150 to 4.883) and the Society of Thoracic Surgeons-European Association for Cardio-Thoracic Surgery score category 2 (OR: 4.373; 95% CI: 1.090 to 29.150) and category 5 (OR: 10.385; 95% CI: 1.717 to 89.016) were significantly related to epilepsy occurrence. Of the 41 patients with epilepsy, 15 (including nine with hypoplastic left heart syndrome) had focal impaired awareness seizures specified as autonomic seizures with vomiting, which tends to escape detection. CONCLUSIONS: We clarified the risk factors for developing epilepsy in children with CHD. We also found that autonomic seizure with vomiting is an important symptom in these children.

DOI： 10.1016/j.pediatrneurol.2023.07.004

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結節性硬化症を背景とした焦点てんかんの生後2ヵ月児に対する早期てんかん外科治療の1例

竹中暁, 品川穣, 秋山麻里, 柴田敬, 土屋弘樹, 秋山倫之, 佐々木達也, 伊達勲, 小林勝弘

脳と発達 55 ( Suppl. ) S423 - S423 2023年5月

皮膚所見を伴い鑑別に苦慮したCerebral proliferative angiopathyの男児例

塚原理恵, 柴田敬, 土屋弘樹, 春間純, 竹内章人, 井上拓志, 石黒友也, 小林勝弘

脳と発達 55 ( Suppl. ) S389 - S389 2023年5月

脳梁離断術後の活動性低下,食思不振が長期化しうる要因とは

土屋弘樹, 大野友香子, 道上理絵, 柴田敬, 秋山麻里, 佐々木達也, 伊達勲, 秋山倫之, 小林勝弘

脳と発達 55 ( Suppl. ) S281 - S281 2023年5月

大家族における発達性ディスレクシアの病因遺伝子解析

岡牧郎, 禅正和真, 柴田敬, 宮崎育子, 浅沼幹人, 小林勝弘

脳と発達 55 ( Suppl. ) S353 - S353 2023年5月

脳梁離断術後の活動性低下,食思不振が長期化しうる要因とは

土屋弘樹, 大野友香子, 道上理絵, 柴田敬, 秋山麻里, 佐々木達也, 伊達勲, 秋山倫之, 小林勝弘

脳と発達 55 ( Suppl. ) S281 - S281 2023年5月

結節性硬化症を背景とした焦点てんかんの生後2ヵ月児に対する早期てんかん外科治療の1例

竹中暁, 品川穣, 秋山麻里, 柴田敬, 土屋弘樹, 秋山倫之, 佐々木達也, 伊達勲, 小林勝弘

脳と発達 55 ( Suppl. ) S423 - S423 2023年5月

皮膚所見を伴い鑑別に苦慮したCerebral proliferative angiopathyの男児例

塚原理恵, 柴田敬, 土屋弘樹, 春間純, 竹内章人, 井上拓志, 石黒友也, 小林勝弘

脳と発達 55 ( Suppl. ) S389 - S389 2023年5月

Comprehensive study of metabolic changes induced by a ketogenic diet therapy using GC/MS- and LC/MS-based metabolomics 査読

Mari Akiyama, Tomoyuki Akiyama, Daisuke Saigusa, Eiji Hishinuma, Naomi Matsukawa, Takashi Shibata, Hiroki Tsuchiya, Atsushi Mori, Yuji Fujii, Yukiko Mogami, Chiho Tokorodani, Kozue Kuwahara, Yurika Numata-Uematsu, Kenji Inoue, Katsuhiro Kobayashi

Seizure 107 52 - 59 2023年4月

【てんかん診療update-診断,治療,社会整備,基礎研究の最新情報-】デジタル脳波がもたらす新時代

小林勝弘, 柴田敬, 土屋弘樹

日本臨床 80 ( 12 ) 1895 - 1899 2022年12月

Artificial intelligence-based detection of epileptic discharges from pediatric scalp electroencephalograms: a pilot study 査読

Katsuhiro Kobayashi, Takashi Shibata, Hiroki Tsuchiya, Tomoyuki Akiyama

Acta Medica Okayama 76 ( 6 ) 617 - 624 2022年12月

記述言語：英語掲載種別：研究論文（学術雑誌）

We developed an artificial intelligence (AI) technique to identify epileptic discharges (spikes) in pediatric scalp electroencephalograms (EEGs). We built a convolutional neural network (CNN) model to automatically classify steep potential images into spikes and background activity. For the CNN model' training and validation, we examined 100 children with spikes in EEGs and another 100 without spikes. A different group of 20 children with spikes and 20 without spikes were the actual test subjects. All subjects were ≥ 3 to < 18 years old. The accuracy, sensitivity, and specificity of the analysis were >0.97 when referential and combination EEG montages were used, and < 0.97 with a bipolar montage. The correct classification of background activity in individual patients was significantly better with a referential montage than with a bipolar montage (p=0.0107). Receiver operating characteristic curves yielded an area under the curve > 0.99, indicating high performance of the classification method. EEG patterns that interfered with correct classification included vertex sharp transients, sleep spindles, alpha rhythm, and low-amplitude ill-formed spikes in a run. Our results demonstrate that AI is a promising tool for automatically interpreting pediatric EEGs. Some avenues for improving the technique were also indicated by our findings.

DOI： 10.18926/AMO/64111

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