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伴 SCN1A 突变的 GEFS+相关性颞顶枕部癫痫。

Temporal-parietal-occipital epilepsy in GEFS+ associated with SCN1A mutation.

机构信息

Pediatric Neurology and Muscular Diseases Unit, IRCCS "G. Gaslini" Institute, Genova, Italy.

Department of Neuroscience, Reproductive and Odontostomatological Sciences, Federico II University, Napoli, Italy.

出版信息

Epileptic Disord. 2021 Apr 1;23(2):397-401. doi: 10.1684/epd.2021.1266.

Abstract

Most families with genetic epilepsy with febrile seizures plus show a mutation in the sodium channel alpha 1 subunit gene, however, but there is much phenotypic heterogeneity and focal epilepsy remains relatively rare. Here, we report a family with electroclinical features indicative of temporal-parietal-occipital carrefour epilepsy with common occurrence of post-ictal migraine. We studied a four-generation family including nine affected subjects by means of EEG and MRI. Genetic testing was performed by targeted re-sequencing (gene panel). In most patients, seizure semiology included cognitive, autonomic, and emotional symptoms, eventually evolving towards sensory visual phenomena. Focal sensory vestibular seizures and changes in body perception were also reported in some cases. Post-ictal migraine was common, occurring in five out of the six (83%) epilepsy patients. A missense mutation (c.1130 G>A; p.R377Q) affecting the S5-S6 segment (pore region) of the sodium channel alpha 1 subunit was identified in all affected and four unaffected subjects. Temporal-parietal-occipital carrefour epilepsy is part of the genetic epilepsy with febrile seizures plus spectrum. The electroclinical features in this family support the involvement of a genetically impaired neural network. High prevalence of post-ictal migraine suggests the role of posterior brain areas in the clinical expression of this gene defect.

摘要

大多数伴有发热性惊厥附加症的遗传性癫痫家族显示钠离子通道α 1 亚基基因突变,但存在明显的表型异质性,局灶性癫痫仍然相对少见。在此,我们报告一个具有颞顶枕交界性癫痫特征的家族,其常见的癫痫发作后偏头痛。我们通过脑电图和 MRI 研究了一个四代共 9 名受累者的家族。通过靶向重测序(基因 panel)进行了基因检测。在大多数患者中,发作的症状学包括认知、自主和情感症状,最终发展为感觉视觉现象。在一些病例中,还报告了局灶性感觉前庭发作和身体感知的改变。癫痫发作后偏头痛很常见,6 例(83%)癫痫患者中有 5 例发生。在所有受累者和 4 名未受累者中均发现了影响钠离子通道α 1 亚基 S5-S6 节段(孔区)的错义突变(c.1130 G>A;p.R377Q)。颞顶枕交界性癫痫是伴发热性惊厥附加症的遗传性癫痫谱的一部分。该家族的电临床特征支持受遗传影响的神经网络的参与。癫痫发作后偏头痛的高患病率提示后部脑区在该基因缺陷的临床表现中的作用。

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