Department of Pediatrics, University of California, San Francisco, California 94143-0706, USA.
Am J Med Genet A. 2010 Oct;152A(10):2574-7. doi: 10.1002/ajmg.a.33596.
We report on the occurrence of congenital diaphragmatic hernia in a family with craniofrontonasal syndrome found to have a previously unreported mutation in EFNB1. The female proband presented with hypertelorism, telecanthus, bifid nasal tip, widow's peak, frontal bossing, and a widened metopic suture. Her father was noted to have hypertelorism, telecanthus, widow's peak, and a history of pectus carinatum. He was found to have a previously unreported mutation in exon 5 of EFNB1 predicted to cause premature protein truncation. The parents of the proband previously had a female fetus with congenital diaphragmatic hernia. The occurrence of congenital diaphragmatic hernia, phenotypic differences between males and females, and utility of molecular testing in craniofrontonasal syndrome are demonstrated.
我们报告了一个颅额鼻面综合征家族中发生先天性膈疝的情况,该家族发现 EFNB1 中存在一个以前未报道的突变。女性先证者表现为眼球突出、内眦赘皮、鼻端分叉、尖颅、额骨突出和额缝增宽。她的父亲被发现有眼球突出、内眦赘皮、尖颅和鸡胸的病史。他被发现 EFNB1 外显子 5 中有一个以前未报道的突变,预测会导致蛋白质过早截断。先证者的父母之前有一个患有先天性膈疝的女性胎儿。本研究展示了先天性膈疝的发生、男女表型差异以及分子检测在颅额鼻面综合征中的应用。