lenox - gastaut综合征(scn2a相关)的神经病理表现:1例报告及文献复习。

Conner Thompson, Katsiaryna Khatskevich, Cynthia T Welsh, Tiffany G Baker, Daniel C Butler
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引用次数: 0

摘要

lenox - gastaut综合征是一种由多种病因引起的严重儿童癫痫,包括结构异常、影响大脑的炎症、恶性肿瘤、损伤或未知原因。钠通道病变与多种癫痫发作有关,包括lenox - gastaut综合征,因为它们在大脑中的动作电位传播中起作用。SCN2A是一种主要存在于中枢神经系统的电压门控钠通道。我们报告了一例患有lenox - gastaut综合征和杂合SCN2A变异的6岁女性,她在有人监督的沐浴中变得无反应,后来死亡。在其他肉眼和显微镜下的异常中,神经病理学检查显示齿状-橄榄发育不良。我们提出这个病例,强调lenox - gastaut综合征与通道病变(SCN2A变异)相关的罕见神经病理学发现,以及先前报道的SCN2A变异患者脑异常的文献综述。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Neuropathologic Findings in Lennox-Gastaut Syndrome (SCN2A-Related): A Case Report and Review of Literature.

Lennox-Gastaut syndrome is a form of severe childhood epilepsy caused by a variety of etiologies including structural abnormalities, inflammatory conditions affecting the brain, malignancy, injury, or unknown causes. Sodium channelopathies have been linked to multiple seizure disorders, including Lennox-Gastaut syndrome, due to their role in action potential propagation in the brain. SCN2A is one such voltage gated sodium channel found primarily in the central nervous system. We present a case of a 6-year-old female with Lennox-Gastaut syndrome and a heterozygous SCN2A variant who became unresponsive during a supervised bath and later died. The neuropathologic exam was remarkable for dentato-olivary dysplasia, among other gross and microscopic abnormalities. We present this case highlighting rarely documented neuropathologic findings in Lennox-Gastaut syndrome associated with a channelopathy (SCN2A variant) as well as a review of literature of previously reported brain abnormalities in patients with SCN2A variants.

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