Moerman P, Fryns J P
Department of Pathology, Katholieke Universiteit Leuven, Belgium.
Am J Med Genet. 1996 Jun 14;63(3):479-81. doi: 10.1002/(SICI)1096-8628(19960614)63:3<479::AID-AJMG12>3.0.CO;2-J.
We describe a mother with manifestations most consistent with the Rapp-Hodgkin type of ectodermal dysplasia and her malformed newborn son with ectrodactyly, ectodermal dysplasia, cleft palate, and bilateral cystic and obstructive ureteroceles with hydroureters and cystic renal dysplasia as described in the EEC syndrome. This observation suggests that the Rapp-Hodgkin type of ectodermal dysplasia and EEC syndrome, both defined as autosomal dominant conditions with variable expression, may be manifestations of the same mutated gene. We also want to emphasize that urogenital anomaly is another hallmark of the EEC syndrome.
我们描述了一位母亲,其表现与拉普-霍奇金型外胚层发育不良最为相符,以及她患有畸形的新生儿儿子,该儿子患有缺指(趾)畸形、外胚层发育不良、腭裂,还有如EEC综合征中所描述的双侧囊性及梗阻性输尿管囊肿伴肾盂积水和囊性肾发育不良。这一观察结果表明,拉普-霍奇金型外胚层发育不良和EEC综合征,两者均被定义为具有可变表达的常染色体显性疾病,可能是同一突变基因的表现形式。我们还想强调泌尿生殖系统异常是EEC综合征的另一个标志。