一名患有罕见无义突变变异型索托斯综合征的菲律宾男性在接受卡马西平单药治疗后出现睡眠时电癫痫状态。

IF 1.6 4区 生物学 Q4 BIOCHEMISTRY & MOLECULAR BIOLOGY
Journal of Plant Biochemistry and Biotechnology Pub Date : 2024-03-15 eCollection Date: 2024-01-01 DOI:10.47895/amp.vi0.3013
Jeffrey I Lappay, Benilda C Sanchez-Gan, Michelle E Abadingo
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引用次数: 0

摘要

睡眠中电癫痫状态(ESES)是一种与特定遗传疾病、脑部畸形和服用某些抗癫痫药物有关的电图模式。本病例报告旨在介绍服用卡马西平的索托斯综合征(SoS)患者的 ESES 治疗情况。一名九岁的菲律宾男性患者在一岁时出现发热性癫痫发作,临床特征提示为生长过度综合征。头颅影像学检查显示有透明隔腔、胼胝体发育不良和脑室肥大。他从三岁开始接受卡马西平单药治疗。九岁时在慢波睡眠中记录到近乎连续的弥漫性棘波放电,因此转为丙戊酸治疗。随访研究显示,睡眠中出现局灶性癫痫样放电,ESES消失。下一代测序结果显示,核受体结合集域蛋白1的罕见无义突变呈阳性,确诊为SoS。先进的分子遗传学有助于确定 ESES 的病因。迄今为止,这是有据可查的第一例SoS发展为ESES的病例。无论是固有的遗传倾向还是药物诱导,我们都建议避免使用卡马西平,并将丙戊酸作为一线治疗药物。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Electrical Status Epilepticus during Sleep in a Male Filipino with Rare Nonsense Mutation Variant of Sotos Syndrome on Carbamazepine Monotherapy.

Electrical status epilepticus during sleep (ESES) is an electrographic pattern associated with specific genetic disorders, brain malformations, and use of some antiseizure medications. This case report aims to present the management of ESES in Sotos syndrome (SoS) on carbamazepine. A nine-year-old Filipino male with clinical features suggestive of overgrowth syndrome presented with febrile seizure at one year old. Cranial imaging showed cavum septum pellucidum, corpus callosal dysgenesis, and ventriculomegaly. He was on carbamazepine monotherapy starting at three years old. A near continuous diffuse spike-wave discharges in slow wave sleep was recorded at nine years old hence shifted to valproic acid. Follow-up study showed focal epileptiform discharges during sleep with disappearance of ESES. Next generation sequencing tested positive for rare nonsense mutation of nuclear receptor binding set-domain protein 1 confirming the diagnosis of SoS. Advanced molecular genetics contributed to determination of ESES etiologies. To date, this is the first documented case of SoS developing ESES. Whether an inherent genetic predisposition or drug-induced, we recommend the avoidance of carbamazepine and use of valproic acid as first-line therapy.

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来源期刊
Journal of Plant Biochemistry and Biotechnology
Journal of Plant Biochemistry and Biotechnology 生物-生化与分子生物学
CiteScore
3.90
自引率
0.00%
发文量
59
审稿时长
>12 weeks
期刊介绍: The Journal publishes review articles, research papers, short communications and commentaries in the areas of plant biochemistry, plant molecular biology, microbial and molecular genetics, DNA finger printing, micropropagation, and plant biotechnology including plant genetic engineering, new molecular tools and techniques, genomics & bioinformatics.
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