Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue.
Interferon regulatory factor 6 (IRF6) encodes a highly conserved helix-turn-helix DNA binding protein and is a member of the interferon regulatory family of DNA transcription factors. Mutations in IRF6 lead to isolated and syndromic forms of cleft lip and palate, most notably Van der Woude syndrome...
| Main Authors: | , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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Public Library of Science (PLoS)
2013-01-01
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| Series: | PLoS ONE |
| Online Access: | http://europepmc.org/articles/PMC3579850?pdf=render |
| _version_ | 1818583005678010368 |
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| author | Steven Goudy Peggi Angel Britni Jacobs Cynthia Hill Veronica Mainini Arianna L Smith Youssef A Kousa Richard Caprioli Lawrence S Prince Scott Baldwin Brian C Schutte |
| author_facet | Steven Goudy Peggi Angel Britni Jacobs Cynthia Hill Veronica Mainini Arianna L Smith Youssef A Kousa Richard Caprioli Lawrence S Prince Scott Baldwin Brian C Schutte |
| author_sort | Steven Goudy |
| collection | DOAJ |
| description | Interferon regulatory factor 6 (IRF6) encodes a highly conserved helix-turn-helix DNA binding protein and is a member of the interferon regulatory family of DNA transcription factors. Mutations in IRF6 lead to isolated and syndromic forms of cleft lip and palate, most notably Van der Woude syndrome (VWS) and Popliteal Ptyerigium Syndrome (PPS). Mice lacking both copies of Irf6 have severe limb, skin, palatal and esophageal abnormalities, due to significantly altered and delayed epithelial development. However, a recent report showed that MCS9.7, an enhancer near Irf6, is active in the tongue, suggesting that Irf6 may also be expressed in the tongue. Indeed, we detected Irf6 staining in the mesoderm-derived muscle during development of the tongue. Dual labeling experiments demonstrated that Irf6 was expressed only in the Myf5+ cell lineage, which originates from the segmental paraxial mesoderm and gives rise to the muscles of the tongue. Fate mapping of the segmental paraxial mesoderm cells revealed a cell-autonomous Irf6 function with reduced and poorly organized Myf5+ cell lineage in the tongue. Molecular analyses showed that the Irf6-/- embryos had aberrant cytoskeletal formation of the segmental paraxial mesoderm in the tongue. Fate mapping of the cranial neural crest cells revealed non-cell-autonomous Irf6 function with the loss of the inter-molar eminence. Loss of Irf6 function altered Bmp2, Bmp4, Shh, and Fgf10 signaling suggesting that these genes are involved in Irf6 signaling. Based on these data, Irf6 plays important cell-autonomous and non-cell-autonomous roles in muscular differentiation and cytoskeletal formation in the tongue. |
| first_indexed | 2024-12-16T07:58:24Z |
| format | Article |
| id | doaj.art-c1d2ac7c32af420c8d9782a740c42130 |
| institution | Directory Open Access Journal |
| issn | 1932-6203 |
| language | English |
| last_indexed | 2024-12-16T07:58:24Z |
| publishDate | 2013-01-01 |
| publisher | Public Library of Science (PLoS) |
| record_format | Article |
| series | PLoS ONE |
| spelling | doaj.art-c1d2ac7c32af420c8d9782a740c421302022-12-21T22:38:40ZengPublic Library of Science (PLoS)PLoS ONE1932-62032013-01-0182e5627010.1371/journal.pone.0056270Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue.Steven GoudyPeggi AngelBritni JacobsCynthia HillVeronica MaininiArianna L SmithYoussef A KousaRichard CaprioliLawrence S PrinceScott BaldwinBrian C SchutteInterferon regulatory factor 6 (IRF6) encodes a highly conserved helix-turn-helix DNA binding protein and is a member of the interferon regulatory family of DNA transcription factors. Mutations in IRF6 lead to isolated and syndromic forms of cleft lip and palate, most notably Van der Woude syndrome (VWS) and Popliteal Ptyerigium Syndrome (PPS). Mice lacking both copies of Irf6 have severe limb, skin, palatal and esophageal abnormalities, due to significantly altered and delayed epithelial development. However, a recent report showed that MCS9.7, an enhancer near Irf6, is active in the tongue, suggesting that Irf6 may also be expressed in the tongue. Indeed, we detected Irf6 staining in the mesoderm-derived muscle during development of the tongue. Dual labeling experiments demonstrated that Irf6 was expressed only in the Myf5+ cell lineage, which originates from the segmental paraxial mesoderm and gives rise to the muscles of the tongue. Fate mapping of the segmental paraxial mesoderm cells revealed a cell-autonomous Irf6 function with reduced and poorly organized Myf5+ cell lineage in the tongue. Molecular analyses showed that the Irf6-/- embryos had aberrant cytoskeletal formation of the segmental paraxial mesoderm in the tongue. Fate mapping of the cranial neural crest cells revealed non-cell-autonomous Irf6 function with the loss of the inter-molar eminence. Loss of Irf6 function altered Bmp2, Bmp4, Shh, and Fgf10 signaling suggesting that these genes are involved in Irf6 signaling. Based on these data, Irf6 plays important cell-autonomous and non-cell-autonomous roles in muscular differentiation and cytoskeletal formation in the tongue.http://europepmc.org/articles/PMC3579850?pdf=render |
| spellingShingle | Steven Goudy Peggi Angel Britni Jacobs Cynthia Hill Veronica Mainini Arianna L Smith Youssef A Kousa Richard Caprioli Lawrence S Prince Scott Baldwin Brian C Schutte Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue. PLoS ONE |
| title | Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue. |
| title_full | Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue. |
| title_fullStr | Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue. |
| title_full_unstemmed | Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue. |
| title_short | Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue. |
| title_sort | cell autonomous and non cell autonomous roles for irf6 during development of the tongue |
| url | http://europepmc.org/articles/PMC3579850?pdf=render |
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