scn1a相关的发育性和癫痫性脑病的临床和分子特征的新研究

IF 0.1 Q4 CLINICAL NEUROLOGY
Elżbieta Stawicka, P. Górka-Skoczylas, D. Hoffman-Zacharska
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引用次数: 0

摘要

scn1a相关疾病是一组异质性的疾病,具有不断扩大的表型谱。直到最近,该基因的突变与癫痫综合征和癫痫性和发展性脑病-Dravet综合征有关,这与一组新的早发综合征,非Dravet发育性和癫痫性脑病(dee;人类:PS308350)。本文的目的是回顾已发表的关于scn1a相关的发育性和癫痫性脑病,特别是非德拉韦综合征的表型变异性的数据。这些疾病具有非常早发,多态,耐药癫痫发作,精神运动发育受损和智力残疾,以及存在其他症状,如关节挛缩,骨质减少和多动运动障碍。与德拉韦综合征不同,癫痫发作开始于生命的最初几个月,可能有癫痫痉挛或强直性形态。快速识别非dravet发育性和癫痫性脑病的能力具有重要的临床价值,因为致病SCN1A变异的鉴定及其功能评估对治疗和预后都有影响。对非dravet发育性脑病和癫痫性脑病的病因研究表明,在这些患者中发现了功能获得(GOF)型的致病变异。因此,有可能使用钠通道阻滞剂组中的药物治疗这类患者,这些药物在Dravet综合征中发生的功能丧失(LOF)变异病例中是禁忌的。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A new look at the clinical and molecular characteristics of SCN1A-related developmental and epileptic encephalopathies
SCN1A-related diseases are a heterogeneous group of disorders with an expanding spectrum of phenotypes. Until recently, mutations in this gene were associated with epileptic syndromes and epileptic and developmental encephalopathy – Dravet syndrome, which was contrasted with a new group of early-onset syndromes, non-Dravet developmental and epileptic encephalopathies (DEEs; OMIM: PS308350). The aim of this paper is to review published data on the phenotypic variability of SCN1A-related developmental and epileptic encephalopathies, particularly non-Dravet syndromes. These are disorders with very early onset, polymorphic, drug-resistant epileptic seizures, impaired psychomotor development and intellectual disability, as well as the presence of additional symptoms such as arthrogryposis, osteopenia, and hyperkinetic movement disorders. Unlike Dravet syndrome, epileptic seizures begin in the first few months of life and may have an epileptic spasm or tonic morphology. The ability to quickly recognise the non-Dravet developmental and epileptic encephalopathy is of significant clinical value, because the identification of pathogenic SCN1A variants and their functional evaluation have an impact on both treatment and prognosis. Studies on the aetiology of non-Dravet developmental and epileptic encephalopathies have shown that pathogenic variants of the gain of function (GOF) type are identified in these patients. Therefore, it is possible to treat such patients with medicaments from the group of sodium channel blockers, which were contraindicated in cases of loss of function (LOF) variants, occurring in Dravet syndrome.
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来源期刊
Aktualnosci Neurologiczne
Aktualnosci Neurologiczne CLINICAL NEUROLOGY-
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