Ross R S, Bulkley B H, Hutchins G M, Harshey J S, Jones R A, Kraus H, Liebman J, Thorne C M, Weinberg S B, Weech A A, Weech A A
Am Heart J. 1978 Aug;96(2):170-8. doi: 10.1016/0002-8703(78)90082-0.
A peculiar non-hypertrophic myocardiopathy is described which occurred in three and possibly five generations of a single family. Clinical features included systolic murmurs, electrocardiographic abnormalities, and sudden cardiac death with a paucity of symptoms of cardiac dysfunction. Pathological studies in three generations showed a striking similarity of cardiac findings including globular and dilated ventricles, endocardial fibroelastosis, and mitral valve thickening. Myocardium in two showed basophilic degeneration and fibrosis. A retrospective genealogic analysis and a prospective clinical evaluation of living family members suggested an autosomal dominant mode of inheritance with variable penetrance. The cause of this heritable myocardiopathy is presumably a mutant gene; the biochemical defect to which the mutant gene gives rise remains unknown.
本文描述了一种特殊的非肥厚性心肌病,它出现在一个家族的三代人身上,可能还有五代人。临床特征包括收缩期杂音、心电图异常以及心脏功能障碍症状较少的心脏性猝死。三代人的病理学研究显示心脏表现有显著相似性,包括心室球形扩张、心内膜纤维弹性组织增生以及二尖瓣增厚。其中两人的心肌显示嗜碱性变性和纤维化。对在世家庭成员的回顾性系谱分析和前瞻性临床评估表明,这是一种具有可变外显率的常染色体显性遗传模式。这种遗传性心肌病的病因可能是一个突变基因;该突变基因所导致的生化缺陷尚不清楚。