Cardiology Department, The Second Affiliated Hospital of Nanchang University, Nanchang, China.
Eur J Hum Genet. 2012 Nov;20(11):1189-92. doi: 10.1038/ejhg.2012.63. Epub 2012 Apr 11.
Mutations in the α-subunit of cardiac sodium channel gene SCN5A can lead to the overlapping phenotypes of both the Brugada and type 3 long QT syndromes. However, the combination of Brugada and a short QT phenotype resulting from mutation in SCN5A has not previously been described. A man with concomitant Brugada-like and short QT electrocardiogram (ECG) was identified and the SCN5A gene was sequenced. Whole-cell patch clamp analysis of human embryo kidney (HEK) 293 cells expressing a SCN5A channel with the patient's sequence was used to investigate the biophysical properties of the channel. The patient with the family history of sudden death showed Brugada-like and short QT interval ECG. Sequence analysis of the coding region of the SCN5A gene, identified a G to A heterozygous missense mutation at nucleotide site 2066 that resulted in a amino-acid substitution of arginine to histidine at amino-acid site 689 (R689H). Patch clamp analysis showed that the R689H failed to generate current when heterologously expressed in HEK293 cells, indicating it was a loss-of-function mutation. Our finding firstly shows that a heterozygous missense mutation R689H in SCN5A gene results in the loss of protein function and the coexistents of the Brugada-like and short QT interval ECG phenotypes.
心脏钠离子通道基因 SCN5A 的 α 亚单位突变可导致 Brugada 综合征和 3 型长 QT 综合征的重叠表型。然而,以前尚未描述过 SCN5A 突变导致 Brugada 样和短 QT 表型的组合。本研究鉴定了一位同时存在 Brugada 样和短 QT 心电图(ECG)的男性,并对 SCN5A 基因进行了测序。使用表达具有患者序列的 SCN5A 通道的人胚肾(HEK)293 细胞的全细胞膜片钳分析,研究了通道的生物物理特性。具有家族性猝死史的患者表现出 Brugada 样和短 QT 间期 ECG。SCN5A 基因编码区的序列分析,在核苷酸 2066 处发现 G 到 A 杂合错义突变,导致氨基酸 689 处的精氨酸突变为组氨酸(R689H)。膜片钳分析表明,R689H 异源表达于 HEK293 细胞时未能产生电流,表明这是一种失活突变。我们的发现首次表明,SCN5A 基因中的杂合错义突变 R689H 导致蛋白功能丧失,并伴有 Brugada 样和短 QT 间期 ECG 表型共存。