Tan Ene-Choo, Lim Hwee-Woon, Lim Eileen C P, Lee Seng-Teik
Cleft Palate Craniofac J. 2017 Jul;54(4):442-445. doi: 10.1597/15-327. Epub 2016 May 31.
Van der Woude syndrome (VWS) is a rare autosomal dominant genetic disorder characterized by orofacial clefting and lip pits. Mutations in the transcription factor interferon regulatory factor 6 gene (IRF6) have been identified in individuals with VWS. We performed direct sequencing of the gene for molecular investigation of a proband with Bangladeshi-Malay ancestry. A novel transition mutation (c.113T>C), which resulted in an amino acid substitution (p.Ile38Thr) in the deoxyribonucleic acid-binding domain was detected. Testing of family members showed that the mutation segregated with the VWS phenotype for members of her immediate family. Although there is some phenotypic variability, all of the affected members are of the female gender.
范德伍德综合征(VWS)是一种罕见的常染色体显性遗传病,其特征为口面部裂隙和唇凹。在患有VWS的个体中已鉴定出转录因子干扰素调节因子6基因(IRF6)的突变。我们对一名有孟加拉 - 马来血统的先证者进行了该基因的直接测序,以进行分子研究。检测到一个新的转换突变(c.113T>C),该突变导致在脱氧核糖核酸结合域发生氨基酸替换(p.Ile38Thr)。对家庭成员的检测表明,该突变与她直系亲属中的VWS表型共分离。尽管存在一些表型变异性,但所有受影响成员均为女性。