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来自巴基斯坦的范德伍德综合征患者中IRF6的新型唇凹表型和突变

Novel lip pit phenotypes and mutations of IRF6 in Van der Woude syndrome patients from Pakistan.

作者信息

Malik S, Wilcox E R, Naz S

机构信息

School of Biological Sciences; Department of Microbiology and Molecular Genetics, University of the Punjab, Lahore, Pakistan.

出版信息

Clin Genet. 2014 May;85(5):487-91. doi: 10.1111/cge.12207. Epub 2013 Jun 24.

Abstract

The role of interferon regulatory factor 6 (IRF6) gene mutations in causing Van der Woude syndrome (VWS) and poplyteal pterygium syndrome has been described in different populations worldwide. The former is one of the major syndromes of cleft lip and/or cleft palate (CL/P) with the distinct phenotype of presence of pits with or without sinuses on the lower lip. We identified seven probands with VWS from Punjab province of Pakistan and recognized two previously unreported lip pit phenotypes. The mutational analysis of IRF6 in this cohort revealed four novel and two previously reported mutations. The newly identified mutations include three frameshifts (c.635delG; c.21_33del13; c.627delC) and one transition mutation (c.2T>C) affecting the first codon of IRF6. Together with a past epidemiological study on VWS in Pakistan, the frequency of this syndrome among CL/P individuals from Punjab was calculated to be 1.17%.

摘要

干扰素调节因子6(IRF6)基因突变在导致范德伍德综合征(VWS)和腘窝翼状胬肉综合征中的作用已在全球不同人群中得到描述。前者是唇裂和/或腭裂(CL/P)的主要综合征之一,具有下唇有或无窦道的凹陷这一独特表型。我们从巴基斯坦旁遮普省鉴定出7例VWS先证者,并识别出两种先前未报道的唇凹陷表型。对该队列中IRF6的突变分析揭示了4个新突变和2个先前报道的突变。新发现的突变包括3个移码突变(c.635delG;c.21_33del13;c.627delC)和1个影响IRF6第一个密码子的转换突变(c.2T>C)。结合过去在巴基斯坦进行的一项关于VWS的流行病学研究,计算得出旁遮普CL/P个体中该综合征的发生率为1.17%。

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