Siskind Tamar, Williams Nori, Sebastin Monisha, Marion Robert, McDonald Thomas V, Walsh Christine, Sampson Barbara, Tang Yingying, Clark Bradley C
Department of Pediatrics, Children's Hospital at Montefiore, Bronx, New York, NY, USA.
New York City Office of Chief Medical Examiner, New York, NY, USA.
J Community Genet. 2022 Dec;13(6):629-639. doi: 10.1007/s12687-022-00611-1. Epub 2022 Oct 7.
Currently, no standardized system exists for evaluating and testing at-risk family members of decedents with abnormal post-mortem genetic testing in cases of sudden unexpected death (SUD). The goal of this study was to evaluate the outcomes of referrals made by an urban medical examiner's office to a multi-disciplinary cardiogenetics clinic. Relatives of decedents with pathogenic/likely pathogenic (P/LP) variants or variants of unknown significance (VUS) in genes known to be associated with cardiomyopathies and/or arrhythmias were identified by the New York City Office of Chief Medical Examiner and referred to the Cardiogenetics Clinic at Montefiore Medical Center. Familial referrals of 15 decedents (median 15 years, range 2 days to 57 years) were evaluated. Variants in 13 genes were identified among decedents (9 arrhythmia, 5 cardiomyopathy). P/LP variants were identified in both arrhythmia (RYR2, SCN5A) and cardiomyopathy syndrome (MYBPC3 (2), MYH7) genes. Thirty-two family members were referred, and 14 variants were detected. One pathogenic (MYBPC3) and two likely pathogenic (RYR2, MYH7) mutations were identified. Referral of at-risk family members of decedents who experienced SUD based on informative post-mortem genetic testing for cardiac and genetic evaluation is warranted, as family studies help to reclassify variants and prevent additional sudden death.
目前,对于在意外猝死(SUD)病例中尸检基因检测异常的死者的高危家庭成员,不存在用于评估和检测的标准化系统。本研究的目的是评估城市法医办公室转介至多学科心脏遗传学诊所的结果。纽约市首席法医办公室识别出在已知与心肌病和/或心律失常相关的基因中携带致病/可能致病(P/LP)变异或意义未明变异(VUS)的死者亲属,并将其转介至蒙特菲奥里医疗中心的心脏遗传学诊所。对15名死者(年龄中位数15岁,范围2天至57岁)的家族性转介病例进行了评估。在死者中鉴定出13个基因的变异(9个与心律失常相关,5个与心肌病相关)。在心律失常相关基因(RYR2、SCN5A)和心肌病综合征相关基因(MYBPC3(2个)、MYH7)中均鉴定出P/LP变异。共转介了32名家庭成员,检测到14个变异。鉴定出1个致病突变(MYBPC3)和2个可能致病突变(RYR2、MYH7)。基于对心脏和基因评估有参考价值的尸检基因检测,对经历意外猝死的死者的高危家庭成员进行转介是有必要的,因为家族研究有助于对变异进行重新分类并预防更多的猝死发生。