Suppr超能文献

由FZD6突变引起的孤立性隐性甲发育异常:三例家系报告及文献复习

Isolated recessive nail dysplasia caused by FZD6 mutations: report of three families and review of the literature.

作者信息

Kasparis C, Reid D, Wilson N J, Okur V, Cole C, Hansen C D, Bosse K, Betz R C, Khan M, Smith F J D

机构信息

Dermatology Department, Walsall Healthcare NHS Trust, Walsall, UK.

Centre for Dermatology and Genetic Medicine, Division of Biological Chemistry and Drug Discovery, School of Life Sciences, University of Dundee, Dundee, UK.

出版信息

Clin Exp Dermatol. 2016 Dec;41(8):884-889. doi: 10.1111/ced.12934. Epub 2016 Oct 27.

Abstract

Congenital abnormalities of the nail are rare conditions that are most frequently associated with congenital ectodermal syndromes involving several of the epidermal appendages including the skin, teeth, hair and nails. Isolated recessive nail dysplasia (IRND) is much rarer but has recently been recognized as a condition resulting in 20-nail dystrophy in the absence of other cutaneous or extracutaneous findings. A few case reports have identified mutations in the Frizzled 6 (FZD6) gene in families presenting with abnormal nails consistent with IRND. These reports have highlighted the role of Wnt-FZD signalling in the process of nail formation. We report three families presenting with features of IRND, in whom we identified mutations in FZD6, including one previously unreported mutation.

摘要

先天性指甲异常是罕见病症,最常与先天性外胚层综合征相关,该综合征涉及多个表皮附属器,包括皮肤、牙齿、毛发和指甲。孤立性隐性指甲发育不良(IRND)更为罕见,但最近已被确认为一种在无其他皮肤或皮肤外表现的情况下导致20甲营养不良的病症。一些病例报告已在表现出与IRND一致的指甲异常的家族中鉴定出卷曲蛋白6(FZD6)基因的突变。这些报告突出了Wnt-FZD信号在指甲形成过程中的作用。我们报告了三个呈现IRND特征的家族,我们在其中鉴定出了FZD6基因的突变,包括一个先前未报告的突变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9590/5132090/855415f8298b/CED-41-884-g001.jpg

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