The etiology of cleft palate formation in BMP7-deficient mice.

Palatogenesis is a complex process implying growth, elevation and fusion of the two lateral palatal shelves during embryogenesis. This process is tightly controlled by genetic and mechanistic cues that also coordinate the growth of other orofacial structures. Failure at any of these steps can result...

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Autores principales:	Thaleia Kouskoura, Anastasiia Kozlova, Maria Alexiou, Susanne Blumer, Vasiliki Zouvelou, Christos Katsaros, Matthias Chiquet, Thimios A Mitsiadis, Daniel Graf
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2013
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/24a2486e6de447c4835e07d3493e0368
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spelling	oai:doaj.org-article:24a2486e6de447c4835e07d3493e03682021-11-18T07:53:21ZThe etiology of cleft palate formation in BMP7-deficient mice.1932-620310.1371/journal.pone.0059463https://doaj.org/article/24a2486e6de447c4835e07d3493e03682013-01-01T00:00:00Zhttps://www.ncbi.nlm.nih.gov/pmc/articles/pmid/23516636/pdf/?tool=EBIhttps://doaj.org/toc/1932-6203Palatogenesis is a complex process implying growth, elevation and fusion of the two lateral palatal shelves during embryogenesis. This process is tightly controlled by genetic and mechanistic cues that also coordinate the growth of other orofacial structures. Failure at any of these steps can result in cleft palate, which is a frequent craniofacial malformation in humans. To understand the etiology of cleft palate linked to the BMP signaling pathway, we studied palatogenesis in Bmp7-deficient mouse embryos. Bmp7 expression was found in several orofacial structures including the edges of the palatal shelves prior and during their fusion. Bmp7 deletion resulted in a general alteration of oral cavity morphology, unpaired palatal shelf elevation, delayed shelf approximation, and subsequent lack of fusion. Cell proliferation and expression of specific genes involved in palatogenesis were not altered in Bmp7-deficient embryos. Conditional ablation of Bmp7 with Keratin14-Cre or Wnt1-Cre revealed that neither epithelial nor neural crest-specific loss of Bmp7 alone could recapitulate the cleft palate phenotype. Palatal shelves from mutant embryos were able to fuse when cultured in vitro as isolated shelves in proximity, but not when cultured as whole upper jaw explants. Thus, deformations in the oral cavity of Bmp7-deficient embryos such as the shorter and wider mandible were not solely responsible for cleft palate formation. These findings indicate a requirement for Bmp7 for the coordination of both developmental and mechanistic aspects of palatogenesis.Thaleia KouskouraAnastasiia KozlovaMaria AlexiouSusanne BlumerVasiliki ZouvelouChristos KatsarosMatthias ChiquetThimios A MitsiadisDaniel GrafPublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 8, Iss 3, p e59463 (2013)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Thaleia Kouskoura Anastasiia Kozlova Maria Alexiou Susanne Blumer Vasiliki Zouvelou Christos Katsaros Matthias Chiquet Thimios A Mitsiadis Daniel Graf The etiology of cleft palate formation in BMP7-deficient mice.
description	Palatogenesis is a complex process implying growth, elevation and fusion of the two lateral palatal shelves during embryogenesis. This process is tightly controlled by genetic and mechanistic cues that also coordinate the growth of other orofacial structures. Failure at any of these steps can result in cleft palate, which is a frequent craniofacial malformation in humans. To understand the etiology of cleft palate linked to the BMP signaling pathway, we studied palatogenesis in Bmp7-deficient mouse embryos. Bmp7 expression was found in several orofacial structures including the edges of the palatal shelves prior and during their fusion. Bmp7 deletion resulted in a general alteration of oral cavity morphology, unpaired palatal shelf elevation, delayed shelf approximation, and subsequent lack of fusion. Cell proliferation and expression of specific genes involved in palatogenesis were not altered in Bmp7-deficient embryos. Conditional ablation of Bmp7 with Keratin14-Cre or Wnt1-Cre revealed that neither epithelial nor neural crest-specific loss of Bmp7 alone could recapitulate the cleft palate phenotype. Palatal shelves from mutant embryos were able to fuse when cultured in vitro as isolated shelves in proximity, but not when cultured as whole upper jaw explants. Thus, deformations in the oral cavity of Bmp7-deficient embryos such as the shorter and wider mandible were not solely responsible for cleft palate formation. These findings indicate a requirement for Bmp7 for the coordination of both developmental and mechanistic aspects of palatogenesis.
format	article
author	Thaleia Kouskoura Anastasiia Kozlova Maria Alexiou Susanne Blumer Vasiliki Zouvelou Christos Katsaros Matthias Chiquet Thimios A Mitsiadis Daniel Graf
author_facet	Thaleia Kouskoura Anastasiia Kozlova Maria Alexiou Susanne Blumer Vasiliki Zouvelou Christos Katsaros Matthias Chiquet Thimios A Mitsiadis Daniel Graf
author_sort	Thaleia Kouskoura
title	The etiology of cleft palate formation in BMP7-deficient mice.
title_short	The etiology of cleft palate formation in BMP7-deficient mice.
title_full	The etiology of cleft palate formation in BMP7-deficient mice.
title_fullStr	The etiology of cleft palate formation in BMP7-deficient mice.
title_full_unstemmed	The etiology of cleft palate formation in BMP7-deficient mice.
title_sort	etiology of cleft palate formation in bmp7-deficient mice.
publisher	Public Library of Science (PLoS)
publishDate	2013
url	https://doaj.org/article/24a2486e6de447c4835e07d3493e0368
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The etiology of cleft palate formation in BMP7-deficient mice.

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