Ackerman Michael J, Splawski Igor, Makielski Jonathan C, Tester David J, Will Melissa L, Timothy Katherine W, Keating Mark T, Jones Gregg, Chadha Monica, Burrow Christopher R, Stephens J Claiborne, Xu Chuanbo, Judson Richard, Curran Mark E
Mayo Clinic College of Medicine, Rochester, Minnesota 55905, USA.
Heart Rhythm. 2004 Nov;1(5):600-7. doi: 10.1016/j.hrthm.2004.07.013.
The purpose of this study was to determine the prevalence and spectrum of nonsynonymous polymorphisms (amino acid variants) in the cardiac sodium channel among healthy subjects.
Pathogenic mutations in the cardiac sodium channel gene, SCN5A, cause approximately 15 to 20% of Brugada syndrome (BrS1), 5 to 10% of long QT syndrome (LQT3), and 2 to 5% of sudden infant death syndrome.
Using single-stranded conformation polymorphism, denaturing high-performance liquid chromatography, and/or direct DNA sequencing, mutational analysis of the protein-encoding exons of SCN5A was performed on 829 unrelated, anonymous healthy subjects: 319 black, 295 white, 112 Asian, and 103 Hispanic.
In addition to the four known common polymorphisms (R34C, H558R, S1103Y, and R1193Q), four relatively ethnic-specific polymorphisms were identified: R481W, S524Y, P1090L, and V1951L. Overall, 39 distinct missense variants (28 novel) were elucidated. Nineteen variants (49%) were found only in the black cohort. Only seven variants (18%) localized to transmembrane-spanning domains. Four variants (F1293S, R1512W, and V1951L cited previously as BrS1-causing mutations and S1787N previously published as a possible LQT3-causing mutation) were identified in this healthy cohort.
This study provides the first comprehensive determination of the prevalence and spectrum of cardiac sodium channel variants in healthy subjects from four distinct ethnic groups. This compendium of SCN5A variants is critical for proper interpretation of SCN5A genetic testing and provides an essential hit list of targets for future functional studies to determine whether or not any of these variants mediate genetic susceptibility for arrhythmias in the setting of either drugs or disease.
本研究旨在确定健康受试者心脏钠通道非同义多态性(氨基酸变体)的患病率和谱型。
心脏钠通道基因SCN5A中的致病突变导致约15%至20%的Brugada综合征(BrS1)、5%至10%的长QT综合征(LQT3)以及2%至5%的婴儿猝死综合征。
对829名无亲缘关系的匿名健康受试者(319名黑人、295名白人、112名亚洲人和103名西班牙裔)进行了单链构象多态性、变性高效液相色谱和/或直接DNA测序,对SCN5A的蛋白质编码外显子进行突变分析。
除了四个已知的常见多态性(R34C、H558R、S1103Y和R1193Q)外,还鉴定出四个相对种族特异性的多态性:R481W、S524Y、P1090L和V1951L。总体而言,阐明了39个不同的错义变体(28个新变体)。19个变体(49%)仅在黑人队列中发现。只有7个变体(18%)定位于跨膜结构域。在这个健康队列中鉴定出4个变体(F1293S、R1512W,之前被认为是导致BrS1的突变,以及之前发表的可能导致LQT3的突变S1787N)。
本研究首次全面确定了来自四个不同种族的健康受试者心脏钠通道变体的患病率和谱型。这份SCN5A变体汇编对于正确解释SCN5A基因检测至关重要,并为未来功能研究提供了一份重要的目标清单,以确定这些变体是否在药物或疾病背景下介导心律失常的遗传易感性。