Danish National Research Foundation Centre for Cardiac Arrhythmia, Department of Biomedical Sciences, University of Copenhagen, Blegdamsvej 3, DK-2200 Copenhagen N, Denmark.
Can J Physiol Pharmacol. 2011 Oct;89(10):723-36. doi: 10.1139/y11-070. Epub 2011 Sep 6.
Mutations in the SCN5A gene have been linked to Brugada syndrome (BrS), conduction disease, Long QT syndrome (LQT3), atrial fibrillation (AF), and to pre- and neonatal ventricular arrhythmias.
The objective of this study is to characterize a novel mutation in Na(v)1.5 found in a newborn with fetal chaotic atrial tachycardia, post-partum intraventricular conduction delay, and QT interval prolongation.
Genomic DNA was isolated and all exons and intron borders of 15 ion-channel genes were sequenced, revealing a novel missense mutation (Q270K) in SCN5A. Na(v)1.5 wild type (WT) and Q270K were expressed in CHO-K1 with and without the Na(v)β1 subunit. Results. Patch-clamp analysis showed ∼40% reduction in peak sodium channel current (I(Na)) density for Q270K compared with WT. Fast and slow decay of I(Na) were significantly slower in Q270K. Steady-state activation and inactivation of Q270K channels were shifted to positive potentials, and window current was increased. The tetrodotoxin-sensitive late I(Na) was increased almost 3-fold compared with WT channels. Ranolazine reduced late I(Na) in WT and Q270K channels, while exerting minimal effects on peak I(Na).
The Q270K mutation in SCN5A reduces peak I(Na) while augmenting late I(Na), and may thus underlie the development of atrial tachycardia, intraventricular conduction delay, and QT interval prolongation in an infant.
SCN5A 基因突变与 Brugada 综合征(BrS)、传导疾病、长 QT 综合征(LQT3)、心房颤动(AF)以及产前和新生儿室性心律失常有关。
本研究旨在描述一种新发现的 Na(v)1.5 基因突变,该突变存在于一名患有胎儿杂乱性房性心动过速、产后室内传导延迟和 QT 间期延长的新生儿中。
分离基因组 DNA,并对 15 个离子通道基因的所有外显子和内含子边界进行测序,结果显示 SCN5A 中存在一种新的错义突变(Q270K)。在 CHO-K1 细胞中表达 Na(v)1.5 野生型(WT)和 Q270K,并与 Na(v)β1 亚基共表达。结果:膜片钳分析显示,与 WT 相比,Q270K 的峰值钠通道电流(I(Na))密度降低了约 40%。I(Na)的快速和慢速衰减在 Q270K 中明显变慢。Q270K 通道的稳态激活和失活向正电势偏移,窗电流增加。与 WT 通道相比,河豚毒素敏感的晚期 I(Na)增加了近 3 倍。雷诺嗪降低了 WT 和 Q270K 通道中的晚期 I(Na),而对峰值 I(Na)的影响最小。
SCN5A 中的 Q270K 突变降低了峰值 I(Na),同时增加了晚期 I(Na),因此可能导致婴儿出现房性心动过速、室内传导延迟和 QT 间期延长。