Orstavik K H, Kloster R, Lippestad C, Rode L, Hovig T, Fuglseth K N
Department of Medical Genetics, Ullevål Hospital, Oslo, Norway.
Clin Genet. 1990 Dec;38(6):447-51. doi: 10.1111/j.1399-0004.1990.tb03611.x.
Emery-Dreifuss syndrome is characterized by early contractures, slowly progressing muscle wasting and cardiomyopathy, often presenting as heart block. The syndrome is usually inherited as an X-linked recessive. We present a family with four affected females in three generations, including a pair of identical twins. All patients developed elbow contractures, scoliosis, and stiffness of the spine and neck from the age of about 10, with little progression in later years. The proband developed cardiomyopathy at the age of 45, whereas her mother died at 41 without a confirmed diagnosis of cardiomyopathy. The twin daughters of the proband had no unequivocal signs of cardiomyopathy at the age of 21 years. Early recognition of this syndrome is important because of the possible development of heart block.
埃默里-德赖富斯综合征的特征为早期挛缩、缓慢进展的肌肉萎缩和心肌病,常表现为心脏传导阻滞。该综合征通常为X连锁隐性遗传。我们报告一个三代中有四名受累女性的家族,其中包括一对同卵双胞胎。所有患者从大约10岁起出现肘部挛缩、脊柱侧弯以及脊柱和颈部僵硬,在随后几年进展甚微。先证者45岁时患心肌病,而她的母亲41岁去世,未确诊心肌病。先证者的双胞胎女儿在21岁时没有明确的心肌病迹象。由于可能发生心脏传导阻滞,早期识别该综合征很重要。