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多基因panel 测序在 KCNQ1、KCNH2 和 SCN5A 中未检测到致病性变异的长校正 QT 间期患者中的应用。

Application of Multigene Panel Sequencing in Patients with Prolonged Rate-corrected QT Interval and No Pathogenic Variants Detected in KCNQ1, KCNH2, and SCN5A.

机构信息

Department of Laboratory Medicine, Seoul National University Bundang Hospital, Seongnam, Korea.

Department of Laboratory Medicine, National Medical Center, Seoul, Korea.

出版信息

Ann Lab Med. 2018 Jan;38(1):54-58. doi: 10.3343/alm.2018.38.1.54.

Abstract

Long QT syndrome (LQTS) is an inherited cardiac disease characterized by a prolonged heart rate-corrected QT (QTc) interval. We investigated the genetic causes in patients with prolonged QTc intervals who were negative for pathogenic variants in three major LQTS-related genes (KCNQ1, KCNH2, and SCN5A). Molecular genetic testing was performed using a panel including 13 LQTS-related genes and 67 additional genes implicated in other cardiac diseases. Overall, putative genetic causes of prolonged QTc interval were identified in three of the 30 patients (10%). Among the LQTS-related genes, we detected a previously reported pathogenic variant, CACNA1C c.1552C>T, responsible for cardiac-only Timothy syndrome. Among the genes related to other cardiac diseases, a likely pathogenic variant, RYR2 c.11995A>G, was identified in a patient with catecholaminergic polymorphic ventricular tachycardia. Another patient who developed dilated cardiomyopathy with prolonged QTc interval was found to carry a likely pathogenic variant, TAZ c.718G>A, associated with infantile dilated cardiomyopathy. Comprehensive screening of genetic variants using multigene panel sequencing enables detection of genetic variants with a possible involvement in QTc interval prolongation, thus uncovering unknown molecular mechanisms underlying LQTS.

摘要

长 QT 综合征(LQTS)是一种遗传性心脏病,其特征是心率校正 QT(QTc)间期延长。我们研究了三个主要的 LQTS 相关基因(KCNQ1、KCNH2 和 SCN5A)中致病性变异阴性且 QTc 间期延长的患者的遗传原因。使用包括 13 个 LQTS 相关基因和 67 个其他与心脏疾病相关的基因的panel 进行分子遗传学检测。总的来说,在 30 名患者中的 3 名(10%)中确定了导致 QTc 间期延长的潜在遗传原因。在 LQTS 相关基因中,我们检测到了一个先前报道的致病性变异 CACNA1C c.1552C>T,它导致了仅心脏的 Timothy 综合征。在与其他心脏疾病相关的基因中,在一名患有儿茶酚胺多形性室性心动过速的患者中发现了一个可能的致病性变异 RYR2 c.11995A>G。另一名患有扩张型心肌病和 QTc 间期延长的患者携带了一个可能的致病性变异 TAZ c.718G>A,该变异与婴儿扩张型心肌病有关。使用多基因panel 测序对遗传变异进行综合筛查,可以检测到可能导致 QTc 间期延长的遗传变异,从而揭示 LQTS 潜在的未知分子机制。

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