Yamagishi H, Furutani M, Kamisago M, Morikawa Y, Kojima Y, Hino Y, Furutani Y, Kimura M, Imamura S, Takao A, Momma K, Matsuoka R
Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan.
Hum Mutat. 1998;11(6):481. doi: 10.1002/(SICI)1098-1004(1998)11:6<481::AID-HUMU12>3.0.CO;2-Q.
Two missense mutations and a nine-nucleotide deletion of the cardiac sodium channel (SCN5A) gene have been shown to cause long QT syndrome (LQTS) in several familial cases. We identified a novel missense mutation (R1623Q) of the SCN5A gene in a Japanese girl with sporadic LQTS. We used polymerase chain reaction, single-strand conformation polymorphism analysis and DNA sequence analysis to identify a mutation of the SCN5A gene in the patient. A single nucleotide substitution of guanine to adenine, in codon 1612, changed the coding sense of the SCN5A from arginine to glutamine (R1623Q) in the S4 segment of domain IV which is a highly conserved region of the SCN5A. This mutation was not identified in the unaffected biological parents and brother of the patient, and 100 normal, unrelated individuals. This finding is the first evidence of a de nova mutation in SCN5A associated with LQTS.
在几例家族性病例中,已证实心脏钠通道(SCN5A)基因的两个错义突变和一个9核苷酸缺失可导致长QT综合征(LQTS)。我们在一名患有散发性LQTS的日本女孩中鉴定出SCN5A基因的一个新的错义突变(R1623Q)。我们使用聚合酶链反应、单链构象多态性分析和DNA序列分析来鉴定该患者SCN5A基因的突变。第1612密码子中的鸟嘌呤单核苷酸替换为腺嘌呤,使SCN5A在结构域IV的S4段(SCN5A的一个高度保守区域)的编码意义从精氨酸变为谷氨酰胺(R1623Q)。在该患者未受影响的亲生父母和兄弟以及100名正常、无亲缘关系的个体中未发现此突变。这一发现是与LQTS相关的SCN5A基因新发突变的首个证据。