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一个家族中与长QT综合征和致心律失常性右心室心肌病相关的新型SCN5A突变的特征分析

Characterization of a novel SCN5A mutation associated with long QT syndrome and arrhythmogenic right ventricular cardiomyopathy in a family.

作者信息

Li Rui, Zheng Da, Lin Chunxi, Chen Yili, Bai Yang, Zhou Nan, Zhao Qianhao, Wei Wenzhao, Wu Qiuping, Deng Jiacheng, Zhao Shuquan, Yao Hui, Tang Shuangbo, Luo Bin, Liu Shuiping, Quan Li, Liu Xiaoshan, Cheng Jianding, Huang Erwen

机构信息

Department of Forensic Pathology, Zhongshan School of Medicine, Sun Yat-sen University, Guangzhou, Guangdong, China.

Guangdong Province Translational Forensic Medicine Engineering Technology Research Center, Sun Yat-sen University, Guangzhou, Guangdong, China.

出版信息

Forensic Sci Med Pathol. 2025 Mar;21(1):33-41. doi: 10.1007/s12024-024-00863-y. Epub 2024 Aug 12.

DOI:10.1007/s12024-024-00863-y
PMID:39133258
Abstract

Sudden cardiac death represents a significant diagnostic challenge for forensic pathologists, particularly in inherited arrhythmia syndromes or cardiomyopathies resulting from genetic defects. Molecular autopsies can reveal the underlying molecular etiology in such cases. In this study, we investigated a family with a history of sudden cardiac death to elucidate the molecular basis responsible for sudden cardiac death. The proband underwent a comprehensive forensic examination. Family members received thorough clinical evaluations, including electrocardiogram, Holter monitoring, echocardiography, and cardiac magnetic imaging. Whole exome sequencing and genetic analysis were performed on the deceased and her parents. In addition, Western blotting and patch-clamp recordings were employed to evaluate the expression and function of the mutant protein in vitro. Forensic examination diagnosed arrhythmogenic right ventricular cardiomyopathy (ARVC) as the cause of sudden death. Genetic analysis identified a novel missense mutation in SCN5A (p.V1323L), which was assessed as likely pathogenic by the ACMG guideline. Another family member carrying the mutation manifested long QT syndrome and mild cardiac fibrosis. The cellular electrophysiological study demonstrated that the mutation resulted in an enhanced late sodium current, suggesting it was a gain-of-function mutation. This study characterizes a novel SCN5A mutation that putatively causes long QT syndrome and may contribute to the development of ARVC. Our work expands the pathogenic spectrum of SCN5A variants and underscores the importance of molecular autopsy in sudden death cases, especially in those with suspected genetic disorders.

摘要

心脏性猝死对法医病理学家来说是一项重大的诊断挑战,尤其是在遗传性心律失常综合征或由基因缺陷导致的心肌病中。分子尸检可以揭示此类病例潜在的分子病因。在本研究中,我们调查了一个有心脏性猝死病史的家族,以阐明导致心脏性猝死的分子基础。先证者接受了全面的法医检查。家庭成员接受了全面的临床评估,包括心电图、动态心电图监测、超声心动图和心脏磁共振成像。对死者及其父母进行了全外显子组测序和基因分析。此外,采用蛋白质免疫印迹法和膜片钳记录来评估突变蛋白在体外的表达和功能。法医检查诊断致心律失常性右室心肌病(ARVC)为猝死原因。基因分析在SCN5A基因中发现了一个新的错义突变(p.V1323L),根据美国医学遗传学与基因组学学会(ACMG)指南,该突变被评估为可能致病。另一名携带该突变的家庭成员表现为长QT综合征和轻度心脏纤维化。细胞电生理研究表明,该突变导致晚期钠电流增强,提示这是一个功能获得性突变。本研究鉴定了一种可能导致长QT综合征并可能促成ARVC发生的新型SCN5A突变。我们的工作扩展了SCN5A变异体的致病谱,并强调了分子尸检在猝死病例中的重要性,特别是在那些疑似遗传性疾病的病例中。

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2
Brugada Syndrome Associated with Different Heterozygous Variants in Two Unrelated Families.两个无关家族中与不同杂合变异相关的Brugada综合征
J Clin Med. 2022 Sep 24;11(19):5625. doi: 10.3390/jcm11195625.
3
Histopathological diagnosis of arrhythmogenic right ventricular cardiomyopathy: A review of three autopsy cases.
心律失常性右室心肌病的组织病理学诊断:三例尸检病例回顾。
Malays J Pathol. 2022 Aug;44(2):277-283.
4
Diagnosis of arrhythmogenic cardiomyopathy: The Padua criteria.心律失常性心肌病的诊断:帕多瓦标准。
Int J Cardiol. 2020 Nov 15;319:106-114. doi: 10.1016/j.ijcard.2020.06.005. Epub 2020 Jun 16.
5
An East Asian Common Variant Vinculin P.Asp841His Was Associated With Sudden Unexplained Nocturnal Death Syndrome in the Chinese Han Population.一种东亚常见的纽蛋白变异体缬氨酸841位点突变为组氨酸与中国汉族人群的不明原因夜间猝死综合征相关。
J Am Heart Assoc. 2017 Apr 3;6(4):e005330. doi: 10.1161/JAHA.116.005330.
6
Multilevel analyses of SCN5A mutations in arrhythmogenic right ventricular dysplasia/cardiomyopathy suggest non-canonical mechanisms for disease pathogenesis.致心律失常性右室发育不良/心肌病中SCN5A突变的多层次分析提示疾病发病机制的非典型机制。
Cardiovasc Res. 2017 Jan;113(1):102-111. doi: 10.1093/cvr/cvw234.
7
Clinical Aspects of Type 3 Long-QT Syndrome: An International Multicenter Study.3型长QT综合征的临床特征:一项国际多中心研究
Circulation. 2016 Sep 20;134(12):872-82. doi: 10.1161/CIRCULATIONAHA.116.021823. Epub 2016 Aug 26.
8
Nanoscale visualization of functional adhesion/excitability nodes at the intercalated disc.闰盘处功能性黏附/兴奋性节点的纳米级可视化。
Nat Commun. 2016 Jan 20;7:10342. doi: 10.1038/ncomms10342.
9
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Circ Res. 2015 Jun 5;116(12):1887-906. doi: 10.1161/CIRCRESAHA.116.304521.
10
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