Merchut M P, Zdonczyk D, Gujrati M
Department of Neurology, Loyola University Medical Center, Maywood, IL 60153.
J Neurol. 1990 Aug;237(5):316-9. doi: 10.1007/BF00314751.
A young woman with humeroperoneal muscular dystrophy and contractures received a heart transplant for a severe dilated cardiomyopathy. Cardiac histopathology consisted of myocyte hypertrophy, interstitial fibrosis, and nuclear hyperchromaticity without mitochondrial abnormalities. Myopathy and heart disease were not clinically evident in her family, although three relatives had unexplained shortened Achilles tendons without weakness. Tendon contractures may be a partial expression of this myopathic disorder, suggesting an autosomal dominant inheritance with variable penetrance. A muscular dystrophy clinically similar to that of the Emery-Dreifuss (EDMD) type can thus occur in women. Rather than the cardiac arrhythmias typical of EDMD, a dilated cardiomyopathy may occur and present with severe congestive heart failure. This is the first report of cardiac transplantation in such a case.
一名患有肱腓型肌营养不良和挛缩的年轻女性因严重扩张型心肌病接受了心脏移植。心脏组织病理学表现为心肌细胞肥大、间质纤维化和核深染,无线粒体异常。她的家族中,肌病和心脏病在临床上并不明显,尽管有三名亲属有不明原因的跟腱缩短但无肌无力。肌腱挛缩可能是这种肌病的部分表现,提示常染色体显性遗传且外显率可变。因此,临床上与埃默里 - 德赖富斯(EDMD)型相似的肌营养不良可发生于女性。与EDMD典型的心律失常不同,可能会出现扩张型心肌病并伴有严重充血性心力衰竭。这是此类病例心脏移植的首例报告。