Suppr超能文献

EFNB1基因致病性突变患者的颅额鼻综合征表型

Phenotypes of craniofrontonasal syndrome in patients with a pathogenic mutation in EFNB1.

作者信息

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

机构信息

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.

出版信息

Eur J Hum Genet. 2014 Aug;22(8):995-1001. doi: 10.1038/ejhg.2013.273. Epub 2013 Nov 27.

Abstract

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.

摘要

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。

相似文献

1
Phenotypes of craniofrontonasal syndrome in patients with a pathogenic mutation in EFNB1.
Eur J Hum Genet. 2014 Aug;22(8):995-1001. doi: 10.1038/ejhg.2013.273. Epub 2013 Nov 27.
2
Aberrant cell segregation in the craniofacial primordium and the emergence of facial dysmorphology in craniofrontonasal syndrome.
PLoS Genet. 2020 Feb 24;16(2):e1008300. doi: 10.1371/journal.pgen.1008300. eCollection 2020 Feb.
4
Report of a family with craniofrontonasal syndrome.
Clin Dysmorphol. 2015 Apr;24(2):79-83. doi: 10.1097/MCD.0000000000000067.
5
Four novel mutations in EFNB1 in Indian patients with craniofrontonasal syndrome.
J Hum Genet. 2019 Sep;64(9):867-873. doi: 10.1038/s10038-019-0638-9. Epub 2019 Jul 8.
7
Craniofrontonasal Syndrome: Atrial Septal Defect With a Novel EFNB1 Gene Mutation.
Cleft Palate Craniofac J. 2015 Mar;52(2):234-6. doi: 10.1597/13-354. Epub 2014 Jun 11.
10
Contiguous gene deletions involving EFNB1, OPHN1, PJA1 and EDA in patients with craniofrontonasal syndrome.
Clin Genet. 2007 Dec;72(6):506-16. doi: 10.1111/j.1399-0004.2007.00905.x. Epub 2007 Oct 16.

引用本文的文献

2
Multi-omic spatial effects on high-resolution AI-derived retinal thickness.
Nat Commun. 2025 Feb 4;16(1):1317. doi: 10.1038/s41467-024-55635-7.
3
Clinical and Genetic Correlation in Neurocristopathies: Bridging a Precision Medicine Gap.
J Clin Med. 2024 Apr 11;13(8):2223. doi: 10.3390/jcm13082223.
4
Fetal MRI of the supratentorial brain abnormalities: what we should know about ventriculomegaly?
Jpn J Radiol. 2023 Dec;41(12):1323-1330. doi: 10.1007/s11604-023-01462-7. Epub 2023 Jul 26.
5
Case report: Craniofrontonasal syndrome caused by a novel variant in the gene in a Colombian woman.
Front Genet. 2023 Jan 4;13:1092301. doi: 10.3389/fgene.2022.1092301. eCollection 2022.
7
Investigating the effects of compound paralogous EPHB receptor mutations on mouse facial development.
Dev Dyn. 2022 Jul;251(7):1138-1155. doi: 10.1002/dvdy.454. Epub 2022 Jan 27.

本文引用的文献

2
A novel de novo mutation within EFNB1 gene in a young girl with craniofrontonasal syndrome.
Cleft Palate Craniofac J. 2012 Jan;49(1):109-13. doi: 10.1597/10-247. Epub 2011 Feb 27.
3
Diverse clinical and genetic aspects of craniofrontonasal syndrome.
Pediatr Neurol. 2011 Feb;44(2):83-7. doi: 10.1016/j.pediatrneurol.2010.10.012.
4
A novel EFNB1 mutation (c.712delG) in a family with craniofrontonasal syndrome and diaphragmatic hernia.
Am J Med Genet A. 2010 Oct;152A(10):2574-7. doi: 10.1002/ajmg.a.33596.
5
The impact of CFNS-causing EFNB1 mutations on ephrin-B1 function.
BMC Med Genet. 2010 Jun 17;11:98. doi: 10.1186/1471-2350-11-98.
6
Craniofrontonasal dysplasia associated with Chiari malformation.
J Neurosurg Pediatr. 2010 Apr;5(4):375-9. doi: 10.3171/2009.10.PEDS09155.
7
Additional EFNB1 mutations in craniofrontonasal syndrome.
Am J Med Genet A. 2008 Aug 1;146A(15):2008-12. doi: 10.1002/ajmg.a.32388.
8
Craniofrontonasal dysplasia: a surgical treatment algorithm.
Plast Reconstr Surg. 2007 Dec;120(7):1943-1956. doi: 10.1097/01.prs.0000287286.12944.9f.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验