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一个中国家族发作性共济失调 2 中发现 CACNA1A 基因的新型无义突变 p.Tyr1957Ter。

Identification of a novel nonsense mutation p.Tyr1957Ter of CACNA1A in a Chinese family with episodic ataxia 2.

机构信息

Department of Neurology, Nanfang Hospital, Southern Medical University, Guangzhou, People's Republic of China.

出版信息

PLoS One. 2013;8(2):e56362. doi: 10.1371/journal.pone.0056362. Epub 2013 Feb 18.

Abstract

Type 2 episodic ataxia (EA2) is the most common subtype among a group of rare hereditary syndromes characterized by recurrent attacks of ataxia. More than 60 mutations and several gene rearrangements due to large deletions in CACNA1A gene have been reported so far for the cause of EA2. Because CACNA1A gene is a large gene containing 47 exons and there is no hot spot mutation, direct sequencing will be a challenge in clinical genetic testing. In this study, we used next generation sequencing technology to identify a novel nonsense mutation of CACNA1A (p.Tyr1957Ter, NP_001120693.1) resulting in truncated protein without 305 amino acids in the c-terminus. Sanger sequencing confirmed the heterozygous mutation of CACNA1A in a Chinese family with 11 affected individuals. Affected individuals experienced recurrent attacks with or without nystagmus, dysarthria, seizure, myokymia, dystonia, weakness, blurred vision, visual field defects, diplopia, migraine, dizziness, nausea and vomiting, sweating and abdominal pain. This is the first report of EA2 in a Chinese family that carries a novel mutation in CACNA1A gene and had abdominal pain as a novel phenotype associated with EA2.

摘要

2 型发作性共济失调(EA2)是一组罕见遗传性综合征中最常见的亚型,其特征是反复发作的共济失调。到目前为止,已经报道了超过 60 种 CACNA1A 基因突变和几种基因重排,这些突变和重排是由于该基因的大片段缺失引起的。由于 CACNA1A 基因是一个包含 47 个外显子的大基因,且没有热点突变,因此直接测序将是临床基因检测中的一个挑战。在本研究中,我们使用下一代测序技术鉴定了 CACNA1A 的一种新的无义突变(p.Tyr1957Ter,NP_001120693.1),导致截短蛋白,其 C 末端缺失 305 个氨基酸。Sanger 测序证实了一个中国家系中 CACNA1A 的杂合突变,该家系中有 11 名受累个体。受累个体经历反复发作,伴有或不伴有眼球震颤、构音障碍、癫痫、肌束震颤、肌张力障碍、无力、视力模糊、视野缺损、复视、偏头痛、头晕、恶心和呕吐、出汗和腹痛。这是首次报道 CACNA1A 基因中的一个新突变导致中国人家系中发生 EA2,且腹痛是与 EA2 相关的一种新的表型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7523/3575407/1a5965e186a3/pone.0056362.g001.jpg

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