Literature DB >> 23713753

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

S Malik1, E R Wilcox, S Naz.   

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%.
© 2013 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  IRF6; Pakistan; Van der Woude syndrome; cleft lip; cleft palate; lip pits

Mesh:

Substances:

Year:  2013        PMID: 23713753     DOI: 10.1111/cge.12207

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  6 in total

Review 1.  Genetics of Cleft Palate and Velopharyngeal Insufficiency.

Authors:  Walter M Sweeney; Steve T Lanier; Chad A Purnell; Arun K Gosain
Journal:  J Pediatr Genet       Date:  2015-03

2.  The prevalence, penetrance, and expressivity of etiologic IRF6 variants in orofacial clefts patients from sub-Saharan Africa.

Authors:  Lord Jephthah Joojo Gowans; Tamara D Busch; Peter A Mossey; Mekonen A Eshete; Wasiu L Adeyemo; Babatunde Aregbesola; Peter Donkor; Fareed K N Arthur; Pius Agbenorku; James Olutayo; Peter Twumasi; Rahman Braimah; Alexander A Oti; Gyikua Plange-Rhule; Solomon Obiri-Yeboah; Fikre Abate; Paa E Hoyte-Williams; Taye Hailu; Jeffrey C Murray; Azeez Butali
Journal:  Mol Genet Genomic Med       Date:  2017-01-12       Impact factor: 2.183

3.  Functional Characterization of a Novel IRF6 Frameshift Mutation From a Van Der Woude Syndrome Family.

Authors:  Mengqi Zhang; Jieni Zhang; Huaxiang Zhao; Vitaly Ievlev; Wenjie Zhong; Wenbin Huang; Robert A Cornell; Jiuxiang Lin; Feng Chen
Journal:  Front Genet       Date:  2020-06-04       Impact factor: 4.599

4.  A clinical and multi‑omics study of Van der Woude syndrome in three generations of a Chinese family.

Authors:  Kai Yang; Xing-Yue Dong; Jue Wu; Jian-Jiang Zhu; Ya Tan; You-Sheng Yan; Li Lin; Dong-Liang Zhang
Journal:  Mol Med Rep       Date:  2020-07-28       Impact factor: 2.952

5.  Novel IRF6 mutations in families with Van Der Woude syndrome and popliteal pterygium syndrome from sub-Saharan Africa.

Authors:  Azeez Butali; Peter A Mossey; Wasiu L Adeyemo; Mekonen A Eshete; LauRen A Gaines; Dee Even; Ramat O Braimah; Babatunde S Aregbesola; Jennifer V Rigdon; Christian I Emeka; Olutayo James; Mobolanle O Ogunlewe; Akinola L Ladeinde; Fikre Abate; Taye Hailu; Ibrahim Mohammed; Paul E Gravem; Milliard Deribew; Mulualem Gesses; Adebowale A Adeyemo; Jeffrey C Murray
Journal:  Mol Genet Genomic Med       Date:  2014-01-27       Impact factor: 2.183

Review 6.  Non-random distribution of deleterious mutations in the DNA and protein-binding domains of IRF6 are associated with Van Der Woude syndrome.

Authors:  Azeez A Alade; Carmen J Buxo-Martinez; Peter A Mossey; Lord J J Gowans; Mekonen A Eshete; Wasiu L Adeyemo; Thirona Naicker; Waheed A Awotoye; Chinyere Adeleke; Tamara Busch; Ada M Toraño; Carolina A Bello; Mairim Soto; Marilyn Soto; Ricardo Ledesma; Myrellis Marquez; Jose F Cordero; Lydia M Lopez-Del Valle; Maria I Salcedo; Natalio Debs; Mary Li; Aline Petrin; Joy Olotu; Colleen Aldous; James Olutayo; Modupe O Ogunlewe; Fekir Abate; Taye Hailu; Ibrahim Muhammed; Paul Gravem; Milliard Deribew; Mulualem Gesses; Mohaned Hassan; John Pape; Oluwole A Adeniyan; Solomon Obiri-Yeboah; Fareed K N Arthur; Alexander A Oti; Olubukola Olatosi; Sara E Miller; Peter Donkor; Martine M Dunnwald; Mary L Marazita; Adebowale A Adeyemo; Jeffrey C Murray; Azeez Butali
Journal:  Mol Genet Genomic Med       Date:  2020-06-17       Impact factor: 2.473

  6 in total

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