Peyrard-Janvid Myriam, Pegelow Marie, Koillinen Hannele, Larsson Catharina, Fransson Ingegerd, Rautio Jorma, Hukki Jyri, Larson Ola, Karsten Agneta L-A, Kere Juha
Department of Biosciences at NOVUM, Karolinska Institutet, Huddinge, Sweden.
Eur J Hum Genet. 2005 Dec;13(12):1261-7. doi: 10.1038/sj.ejhg.5201493.
The interferon regulatory factor 6 gene (IRF6) has been identified as the major Van der Woude (VWS) syndrome and popliteal pterygium (PPS) syndrome gene with mutations in the majority of the kindreds. We have studied altogether 17 kindreds from Sweden, Finland, Norway, Thailand and Singapore, and report here 10 mutations, six of them previously unseen. In two kindreds, we could document de novo mutations, both of them changing a codon for a glutamine residue to a stop. No mutation could be detected in the four VWS kindreds from Finland, suggesting a founder effect for a mutation in an atypical noncoding position. Our findings demonstrate that several distinct mutations occur in the Swedish population, and confirm the general notion of a broad spectrum of IRF6 mutations underlying the VWS/PPS phenotypes.
干扰素调节因子6基因(IRF6)已被确定为主要的范德伍德综合征(VWS)和腘窝翼状胬肉综合征(PPS)基因,大多数家族中都存在该基因突变。我们总共研究了来自瑞典、芬兰、挪威、泰国和新加坡的17个家族,并在此报告10种突变,其中6种是以前未见过的。在两个家族中,我们能够证明是新发突变,这两个突变均将谷氨酰胺残基的密码子变为终止密码子。未在来自芬兰的4个VWS家族中检测到突变,这表明在一个非典型非编码位置的突变存在奠基者效应。我们的研究结果表明,瑞典人群中发生了几种不同的突变,并证实了VWS/PPS表型背后存在广泛的IRF6突变这一普遍观点。