van den Elzen M E P, Twigg S R F, Goos J A C, Hoogeboom A J M, van den Ouweland A M W, Wilkie A O M, Mathijssen I M J
Department of Plastic and Reconstructive Surgery, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
Weatherall Institute of Molecular Medicine, University of Oxford, John Radcliffe Hospital, Oxford, UK.
Eur J Hum Genet. 2014 Aug;22(8):995-1001. doi: 10.1038/ejhg.2013.273. Epub 2013 Nov 27.
Craniofrontonasal syndrome (CFNS) is an X-linked developmental malformation, caused by mutations in the EFNB1 gene, which have only been described since 2004. A genotype-phenotype correlation seems not to be present. As it is of major importance to adequately counsel patients with EFNB1 mutations and their parents, and to improve diagnosis of new patients, more information about the phenotypic features is needed. This study included 23 patients (2 male, 21 female) with confirmed EFNB1 mutations. All patients underwent a thorough physical examination and photographs were taken. If available, radiological images were also consulted. Hypertelorism, longitudinal ridging and/or splitting of nails, a (mild) webbed neck and a clinodactyly of one or more toes were the only consistent features observed in all patients. Frequently observed phenotypic features were bifid tip of the nose (91%), columellar indentation (91%) and low implantation of breasts (90%). In comparison with anthropometric data of facial proportions, patients with CFNS had a significantly different face in multiple respects. An overview of all phenotypic features is shown. Patients with EFNB1 mutations have a clear phenotype. This study will facilitate genetic counseling of parents and patients, and contribute to the diagnostic and screening process of patients with suspected CFNS.
颅额鼻综合征(CFNS)是一种X连锁发育畸形,由EFNB1基因突变引起,该突变自2004年才被描述。似乎不存在基因型与表型的相关性。由于对携带EFNB1突变的患者及其父母进行充分的咨询以及改善对新患者的诊断至关重要,因此需要更多关于表型特征的信息。本研究纳入了23例确诊为EFNB1突变的患者(2例男性,21例女性)。所有患者均接受了全面的体格检查并拍摄了照片。如有可用的影像学图像,也进行了查阅。两眼间距增宽、指甲纵向嵴状隆起和/或裂开、(轻度)蹼颈以及一个或多个脚趾的小指内翻是所有患者中仅有的一致特征。常见的表型特征有鼻尖分叉(91%)、鼻小柱凹陷(91%)和乳房低位附着(90%)。与面部比例的人体测量数据相比,CFNS患者在多个方面面部明显不同。展示了所有表型特征的概述。携带EFNB1突变的患者有明确的表型。本研究将有助于对父母和患者进行遗传咨询,并有助于疑似CFNS患者的诊断和筛查过程。