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年轻人猝死中意义未明的心脏基因变异的尸检解读:一例病例报告及文献综述

The Postmortem Interpretation of Cardiac Genetic Variants of Unknown Significance in Sudden Death in the Young: A Case Report and Review of the Literature.

作者信息

Fadel Saleh, Walker Alfredo E

出版信息

Acad Forensic Pathol. 2020 Dec;10(3-4):166-175. doi: 10.1177/1925362120984868. Epub 2021 Mar 17.

Abstract

Sudden cardiac death (SCD) in adolescents and young adults is a major traumatic event for families and communities. In these cases, it is not uncommon to have a negative autopsy with structurally and histologically normal heart. Such SCD cases are generally attributed to channelopathies, which include long QT syndrome, short QT syndrome, Brugada syndrome, and catecholaminergic polymorphic ventricular tachycardia. Our understanding of the causes for SCDs has changed significantly with the advancements in molecular and genetic studies, where many mutations are now known to be associated with certain channelopathies. Postmortem analysis provides great value in informing decision-making with regard to screening tests and prophylactic measures that should be taken to prevent sudden death in first degree relatives of the decedent. As this is a rapidly advancing field, our ability to identify genetic mutations has surpassed our ability to interpret them. This led to a unique challenge in genetic testing called variants of unknown significance (VUS). VUSs present a diagnostic dilemma and uncertainty for clinicians and patients with regard to next steps. Caution should be exercised when interpreting VUSs since misinterpretation can result in mismanagement of patients and their families. A case of a young adult man with drowning as his proximate cause of death is presented in circumstances where cardiac genetic testing was indicated and undertaken. Eight VUSs in genes implicated in inheritable cardiac dysfunction were identified and the interpretation of VUSs in this scenario is discussed.

摘要

青少年和青年的心脏性猝死(SCD)对家庭和社区来说是一个重大的创伤性事件。在这些案例中,尸检结果显示心脏结构和组织学正常但死因不明的情况并不罕见。这类SCD病例通常归因于离子通道病,包括长QT综合征、短QT综合征、Brugada综合征和儿茶酚胺能多形性室性心动过速。随着分子和基因研究的进展,我们对SCD病因的理解发生了显著变化,现在已知许多突变与某些离子通道病有关。死后分析对于指导筛选测试和采取预防措施以防止死者一级亲属猝死的决策具有重要价值。由于这是一个快速发展的领域,我们识别基因突变的能力已经超过了解释这些突变的能力。这导致了基因检测中一个独特的挑战,即意义未明的变异(VUS)。VUS给临床医生和患者在后续步骤方面带来了诊断困境和不确定性。在解释VUS时应谨慎,因为错误解读可能导致对患者及其家人的管理不当。本文介绍了一例以溺水为直接死因的青年男性病例,该病例进行了心脏基因检测。在与遗传性心脏功能障碍相关的基因中发现了八个VUS,并讨论了在此情况下对VUS的解读。

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