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Dravet综合征中的新型SCN1A变体及评估广泛的患者选择方法。

Novel SCN1A variants in Dravet syndrome and evaluating a wide approach of patient selection.

作者信息

Surovy Milan, Soltysova Andrea, Kolnikova Miriam, Sykora Pavol, Ilencikova Denisa, Ficek Andrej, Radvanszky Jan, Kadasi Ludevit

机构信息

Department of Molecular Biology, Faculty of Natural Sciences, Comenius University, Mlynska dolina Bratislava, 842 15 Slovak Republic.

出版信息

Gen Physiol Biophys. 2016 Jul;35(3):333-42. doi: 10.4149/gpb_2016002. Epub 2016 Apr 5.

Abstract

Voltage-gated sodium channels are essential for generation and propagation of the action potential mainly in nerve and muscle cells. Causative variants in SCN1A gene which codes the main, pore-forming subunit of the channel expressed in central nervous system are associated predominantly with Dravet syndrome (DS), as well as with generalized epilepsy with febrile seizures plus (GEFS+) making it one of the most significant epilepsy gene. Our goal was to determine whether SCN1A screening is relevant in patients with a broad range of epileptic syndromes. 52 patients diagnosed with DS, generalized epilepsy with GEFS+ or similar types of epileptic syndromes were included. Sequencing of the protein coding parts of the gene complemented with MLPA analysis was carried out. One already described nonsense variant, four novel protein truncating variants and a deletion encompassing the whole SCN1A gene were revealed, all in heterozygous state. All identified variants were found in DS patients with 85.7% sensitivity, thus supporting the role of profound SCN1A gene variants in etiology of DS phenotype. No causative variants were identified in any of non-DS epileptic patients in our cohort, suggesting a minor, but not irrelevant role for SCN1A in patients with other types of childhood epilepsy.

摘要

电压门控钠通道对于动作电位的产生和传导至关重要,主要存在于神经和肌肉细胞中。编码中枢神经系统中表达的通道主要成孔亚基的SCN1A基因的致病变异主要与德拉韦特综合征(DS)以及伴有热性惊厥附加症的全身性癫痫(GEFS+)相关,使其成为最重要的癫痫相关基因之一。我们的目标是确定SCN1A基因筛查在广泛癫痫综合征患者中是否具有相关性。纳入了52例诊断为DS、伴有GEFS+的全身性癫痫或类似类型癫痫综合征的患者。对该基因的蛋白质编码部分进行测序,并辅以多重连接依赖探针扩增(MLPA)分析。发现了一个已描述的无义变异、四个新的蛋白质截短变异以及一个包含整个SCN1A基因的缺失,均为杂合状态。所有已鉴定的变异均在DS患者中发现,灵敏度为85.7%,从而支持SCN1A基因深度变异在DS表型病因学中的作用。在我们队列中的任何非DS癫痫患者中均未鉴定出致病变异,这表明SCN1A在其他类型儿童癫痫患者中作用较小,但并非无关紧要。

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