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