Nature Genetics
- 38, 1335 - 1340 (2006)
Published online: 15 October 2006; | doi:10.1038/ng1903
Abnormal skin, limb and craniofacial morphogenesis in mice deficient for interferon regulatory factor 6 (Irf6)Christopher R Ingraham1, Akira Kinoshita1, Shinji Kondo2, Baoli Yang3, Samin Sajan4, Kurt J Trout1, Margaret I Malik1, Martine Dunnwald5, Stephen L Goudy6, Michael Lovett4, Jeffrey C Murray1 & Brian C Schutte11
Department of Pediatrics, University of Iowa, Iowa City, Iowa 52242, USA. 2
Division of Functional Genomics, Center for Frontier Life Sciences, Nagasaki University, Nagasaki, Japan. 3
Department of Obstetrics and Gynecology, University of Iowa, Iowa City, Iowa 52242, USA. 4
Department of Genetics, Washington University School of Medicine, St. Louis, Missouri 63110, USA. 5
Department of Dermatology, University of Iowa, Iowa City, Iowa 52242, USA. 6
Department of Otolaryngology, Vanderbilt University, Nashville, Tennessee 37232, USA.
Correspondence should be addressed to Brian C Schutte brian-schutte@uiowa.edu HoxTbxPaxIRFIrf1Irf2Irf3Irf4Irf5Irf7Irf8Irf9Irf6IRF6Transcription factor paralogs may share a common role in staged or overlapping expression in specific tissues, as in the Hox family. In other cases, family members have distinct roles in a range of embryologic, differentiation or response pathways (as in the Tbx and Pax families). For the interferon regulatory factor (IRF) family of transcription factors, mice deficient in Irf1, Irf2, Irf3, Irf4, Irf5, Irf7, Irf8 or Irf9 have defects in the immune response but show no embryologic abnormalities1,
2,
3,
4,
5,
6,
7. Mice deficient for Irf6 have not been reported, but in humans, mutations in IRF6 cause two mendelian orofacial clefting syndromes8,
9,
10, and genetic variation in IRF6 confers risk for isolated cleft lip and palate11,
12,
13,
14,
15. Here we report that mice deficient for Irf6 have abnormal skin, limb and craniofacial development. Histological and gene expression analyses indicate that the primary defect is in keratinocyte differentiation and proliferation. This study describes a new role for an IRF family member in epidermal development.
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