Ahmad Ferhaan
Department of Genetics, Harvard Medical School, Howard Hughes Medical Institute, Boston, Mass 02115, USA.
Clin Invest Med. 2003 Aug;26(4):167-78.
Arrhythmogenic right ventricular dysplasia or cardiomyopathy (ARVD or ARVC) is an inherited disorder characterized by replacement of the right ventricular myocardium by adipose and fibrous tissue and associated with sudden cardiac death. This disorder may be as prevalent as 6 in 10 000 and causes 12.5%-25% of sudden death events in the young. Nine genetic loci associated with this disease have been ascertained, and mutations in genes at 3 loci have been discovered. These genetic studies have shed light on some of the pathogenetic mechanisms. Mutations in genes encoding desmoplakin and plakoglobin suggest that altered integrity at cardiac myocyte cell-cell junctions may promote myocyte degeneration and death, with the repair process consisting of replacement of myocardium by adipose and fibrous tissue. Mutations in the gene encoding the cardiac ryanodine receptor suggest that cytoplasmic calcium overloading may lead to arrhythmias characteristic of ARVD, and perhaps also the structural changes. Many of the remaining questions concerning the pathogenesis of ARVD can be answered only by the mapping and identification of other genes associated with this disease.
致心律失常性右室发育不良或心肌病(ARVD或ARVC)是一种遗传性疾病,其特征是右心室心肌被脂肪和纤维组织替代,并与心源性猝死相关。这种疾病的发病率可能高达万分之六,在年轻人的心源性猝死事件中占12.5%-25%。已经确定了9个与该疾病相关的基因位点,并且在3个位点的基因中发现了突变。这些基因研究揭示了一些致病机制。编码桥粒斑蛋白和桥粒珠蛋白的基因突变表明,心肌细胞间连接完整性的改变可能促进心肌细胞变性和死亡,修复过程包括脂肪和纤维组织替代心肌。编码心脏雷诺丁受体的基因突变表明,细胞质钙超载可能导致ARVD特有的心律失常,也可能导致结构改变。关于ARVD发病机制的许多其他问题,只有通过绘制和鉴定与该疾病相关的其他基因才能得到解答。