双室性心律失常性心肌病的心源性猝死:MYH6基因的一种新的未描述变体

IF 0.5 Q4 CARDIAC & CARDIOVASCULAR SYSTEMS Cardiogenetics Pub Date : 2023-10-23 DOI:10.3390/cardiogenetics13040014
Pedro Garcia Brás, Isabel Cardoso, José Viegas, Diana Antunes, Sílvia Aguiar Rosa
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引用次数: 0

摘要

心律失常性心肌病(ACM)可能表现为心脏骤停(SCA),证明ACM相关基因的致病变异对其最终诊断至关重要。一位有心脏性猝死家族史的42岁女性患者在两次心脏性猝死流产后被转介到心肌病诊所。在第二次发作中,患者在心肺复苏下转运(停药时间为57分钟),直到植入体外膜氧合。彻底的诊断检查导致双心室ACM的诊断。基因检测显示,ACM患者MYH6基因c.3673G> tp .(Glu 1225*)中存在先前未描述的变异,该变异插入一个过早终止密码子。这被认为是一种可能的致病变异,起源于一个截断的蛋白质,以前在ACM中描述过。患者23岁的女儿MYH6变异呈阳性,心电图异常提示ACM。本病例详细介绍了SCA的复杂鉴别诊断,并探讨了目前双心室ACM诊断的建议。在患有ACM、复发性SCA和SCD家族史的患者中发现MYH6变异似乎支持了MYH6变异在ACM中的致病性假设,其中表型与肌瘤变异的关系尚不清楚。
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Sudden Cardiac Death in Biventricular Arrhythmogenic Cardiomyopathy: A New Undescribed Variant of the MYH6 Gene
Arrhythmogenic cardiomyopathy (ACM) may present with sudden cardiac arrest (SCA), and demonstration of a pathogenic variant in ACM-related genes is crucial for its definitive diagnosis. A 42-year-old female patient with family history of sudden cardiac death (SCD) was referred to the cardiomyopathy clinic after two episodes of aborted SCA. In the second episode, the patient was transported under cardiopulmonary resuscitation (downtime of 57 min) until extracorporeal membrane oxygenation was implanted. A thorough diagnostic work-up led to a diagnosis of biventricular ACM. Genetic testing revealed a previously undescribed variant in ACM patients in the MYH6 gene, c.3673G>T p.(Glu 1225*), which inserts a premature stop codon. This was considered a possible pathogenic variant originating a truncated protein, previously undescribed in ACM. The patient’s 23-year-old daughter was positive for the MYH6 variant and had ECG abnormalities suggestive of ACM. This case details the complex differential diagnosis of SCA and explores the current recommendations for the diagnosis of biventricular ACM. The identification of a MYH6 variant in a patient with ACM, recurrent SCA, and family history of SCD appears to support the hypothesis of the pathogenicity of MYH6 variants in ACM, in which the association of phenotype with sarcomere variants is still unclear.
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来源期刊
Cardiogenetics
Cardiogenetics CARDIAC & CARDIOVASCULAR SYSTEMS-
自引率
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发文量
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审稿时长
11 weeks
期刊最新文献
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