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接受心脏移植和使用左心室辅助装置的终末期心力衰竭患者的基因检测实践与病理评估

Genetic Testing Practices and Pathological Assessments in Patients With End-stage Heart Failure Undergoing Heart Transplantation and Left Ventricular Assist Device Use.

作者信息

Silver Elizabeth, Argiro Alessia, Murray Sarah S, Korty Lauren, Lin Grace, Pretorius Victor, Urey Marcus A, Hong Kimberly N, Adler Eric D, Bui Quan M

机构信息

Division of Cardiovascular Medicine, Department of Medicine, University of California, San Diego, La Jolla, CA, USA.

Cardiomyopathy Unit, University of Florence, Florence, Italy.

出版信息

J Card Fail. 2025 Jul;31(7):1039-1047. doi: 10.1016/j.cardfail.2024.09.015. Epub 2024 Oct 23.

Abstract

BACKGROUND

Genetic cardiomyopathies (CMs) are increasingly recognized as causes of end-stage heart failure (ESHF). Identification of a genetic etiology in ESHF has important prognostic and family implications. However, genetic testing practices are understudied in patients with ESHF.

METHODS

This single-center, retrospective study included consecutive patients with ESHF who underwent heart transplantation (HT) or left ventricular assist device (LVAD) implantation between 2018 and 2023. Data, including genetic testing and pathology reports, were collected from the electronic medical records. Analyses of demographic and clinical characteristics were stratified by genetic-testing completion and the presence of clinically actionable variants. Logistic regression was performed to evaluate for associations between histology findings and genetic variants.

RESULTS

A total of 529 adult patients (mean age 57 years) were included in the study and were predominantly male (79%, 422/529) and non-white (61%, 322/529). Genetic testing was performed in 54% (196/360) of patients with either nonischemic or mixed CMs. A clinically actionable result was identified in 36% (70/196) of patients, of whom only 43% (30/70) had genetic counselor referrals. The most common genetic variants were TTN (32%, 24/75), MYBPC3 (13%, 10/75) and TTR (11%, 8/75). Clinically actionable variants were identified in patients with known heart failure precipitators such as alcohol use. In multivariable analysis, the presence of interstitial fibrosis, specifically diffuse, on pathology was significantly associated with a clinically actionable variant (aOR 2.29, 95% CI [1.08-4.86]; P = 0.03).

CONCLUSION

Patients with ESHF and with nonischemic or mixed CM who were undergoing advanced therapies had low uptakes of genetic services, including testing and counselors, despite high burdens of genetic disease. Pathology findings such as interstitial fibrosis may provide insight into genetic etiology. The underuse of services suggests a need for implementation strategies to improve uptake.

摘要

背景

遗传性心肌病(CMs)越来越被认为是终末期心力衰竭(ESHF)的病因。确定ESHF的遗传病因具有重要的预后和家庭意义。然而,ESHF患者的基因检测实践研究较少。

方法

这项单中心回顾性研究纳入了2018年至2023年间连续接受心脏移植(HT)或左心室辅助装置(LVAD)植入的ESHF患者。从电子病历中收集包括基因检测和病理报告在内的数据。根据基因检测完成情况和临床可操作变异的存在对人口统计学和临床特征进行分析。进行逻辑回归以评估组织学结果与基因变异之间的关联。

结果

该研究共纳入529例成年患者(平均年龄57岁),主要为男性(79%,422/529)和非白人(61%,322/529)。54%(196/360)的非缺血性或混合性CM患者进行了基因检测。36%(70/196)的患者获得了临床可操作结果,其中只有43%(30/70)患者转诊至遗传咨询师处。最常见的基因变异是TTN(32%,24/75)、MYBPC3(13%,10/75)和TTR(11%,8/75)。在已知心力衰竭诱因(如饮酒)的患者中发现了临床可操作变异。在多变量分析中,病理上存在间质纤维化,特别是弥漫性间质纤维化,与临床可操作变异显著相关(调整后比值比2.29,95%置信区间[1.08 - 4.86];P = 0.03)。

结论

尽管遗传疾病负担较高,但接受高级治疗的ESHF且患有非缺血性或混合性CM的患者对包括检测和咨询在内的遗传服务利用率较低。间质纤维化等病理结果可能有助于了解遗传病因。服务利用不足表明需要实施改善利用率的策略。

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