Curran M E, Splawski I, Timothy K W, Vincent G M, Green E D, Keating M T
Department of Human Genetics, University of Utah Health Sciences Center, Salt Lake City 84112.
Cell. 1995 Mar 10;80(5):795-803. doi: 10.1016/0092-8674(95)90358-5.
To identify genes involved in cardiac arrhythmia, we investigated patients with long QT syndrome (LQT), an inherited disorder causing sudden death from a ventricular tachyarrythmia, torsade de pointes. We previously mapped LQT loci on chromosomes 11 (LQT1), 7 (LQT2), and 3 (LQT3). Here, linkage and physical mapping place LQT2 and a putative potassium channel gene, HERG, on chromosome 7q35-36. Single strand conformation polymorphism and DNA sequence analyses reveal HERG mutations in six LQT families, including two intragenic deletions, one splice-donor mutation, and three missense mutations. In one kindred, the mutation arose de novo. Northern blot analyses show that HERG is strongly expressed in the heart. These data indicate that HERG is LQT2 and suggest a likely cellular mechanism for torsade de pointes.
为了鉴定与心律失常相关的基因,我们研究了长QT综合征(LQT)患者,这是一种遗传性疾病,可导致因室性快速心律失常(尖端扭转型室速)而猝死。我们先前已将LQT基因座定位在11号染色体(LQT1)、7号染色体(LQT2)和3号染色体(LQT3)上。在此,连锁分析和物理图谱分析将LQT2及一个假定的钾通道基因HERG定位在7q35 - 36染色体上。单链构象多态性和DNA序列分析揭示了6个LQT家族中的HERG突变,包括2个基因内缺失、1个剪接供体突变和3个错义突变。在一个家族中,该突变是新发的。Northern印迹分析表明HERG在心脏中强烈表达。这些数据表明HERG就是LQT2,并提示了尖端扭转型室速可能的细胞机制。