Literature DB >> 16160700

Novel and de novo mutations of the IRF6 gene detected in patients with Van der Woude or popliteal pterygium syndrome.

Myriam Peyrard-Janvid1, Marie Pegelow, Hannele Koillinen, Catharina Larsson, Ingegerd Fransson, Jorma Rautio, Jyri Hukki, Ola Larson, Agneta L-A Karsten, Juha Kere.   

Abstract

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.

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Year:  2005        PMID: 16160700     DOI: 10.1038/sj.ejhg.5201493

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  5 in total

1.  IRF6 Screening of Syndromic and a priori Non-Syndromic Cleft Lip and Palate Patients: Identification of a New Type of Minor VWS Sign.

Authors:  L Desmyter; M Ghassibe; N Revencu; O Boute; M Lees; G François; C Verellen-Dumoulin; Y Sznajer; A Moncla; H Benateau; K Claes; K Devriendt; M Mathieu; L Van Maldergem; M-C Addor; V Drouin-Garraud; G Mortier; M Bouma; A Dieux-Coeslier; D Genevieve; A Goldenberg; A Gozu; P Makrythanasis; U McEntagart; A Sanchez; C Vilain; S Vermeer; F Connell; J Verheij; S Manouvrier; G Pierquin; S Odent; M Holder-Espinasse; C Vincent-Delorme; Y Gillerot; R Vanwijck; B Bayet; M Vikkula
Journal:  Mol Syndromol       Date:  2010-06-09

2.  Dominant mutations in GRHL3 cause Van der Woude Syndrome and disrupt oral periderm development.

Authors:  Myriam Peyrard-Janvid; Elizabeth J Leslie; Youssef A Kousa; Tiffany L Smith; Martine Dunnwald; Måns Magnusson; Brian A Lentz; Per Unneberg; Ingegerd Fransson; Hannele K Koillinen; Jorma Rautio; Marie Pegelow; Agneta Karsten; Lina Basel-Vanagaite; William Gordon; Bogi Andersen; Thomas Svensson; Jeffrey C Murray; Robert A Cornell; Juha Kere; Brian C Schutte
Journal:  Am J Hum Genet       Date:  2013-12-19       Impact factor: 11.025

3.  Clinical and molecular findings in a Moroccan patient with popliteal pterygium syndrome: a case report.

Authors:  Ilham Ratbi; Nawfal Fejjal; Marie Legendre; Nathalie Collot; Serge Amselem; Abdelaziz Sefiani
Journal:  J Med Case Rep       Date:  2014-12-29

Review 4.  Tooth agenesis and orofacial clefting: genetic brothers in arms?

Authors:  M Phan; F Conte; K D Khandelwal; C W Ockeloen; T Bartzela; T Kleefstra; H van Bokhoven; M Rubini; H Zhou; C E L Carels
Journal:  Hum Genet       Date:  2016-10-03       Impact factor: 4.132

5.  Prevalence and nonrandom distribution of exonic mutations in interferon regulatory factor 6 in 307 families with Van der Woude syndrome and 37 families with popliteal pterygium syndrome.

Authors:  Renata L L Ferreira de Lima; Sarah A Hoper; Michella Ghassibe; Margaret E Cooper; Nicholas K Rorick; Shinji Kondo; Lori Katz; Mary L Marazita; John Compton; Sherri Bale; Ute Hehr; Michael J Dixon; Sandra Daack-Hirsch; Odile Boute; Bénédicte Bayet; Nicole Revencu; Christine Verellen-Dumoulin; Miikka Vikkula; Antônio Richieri-Costa; Danilo Moretti-Ferreira; Jeffrey C Murray; Brian C Schutte
Journal:  Genet Med       Date:  2009-04       Impact factor: 8.822

  5 in total

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