Document Detail

Shox2-deficient mice exhibit a rare type of incomplete clefting of the secondary palate.
MedLine Citation:
PMID:  16141225     Owner:  NLM     Status:  MEDLINE    
The short stature homeobox gene SHOX is associated with idiopathic short stature in humans, as seen in Turner syndrome and Leri-Weill dyschondrosteosis, while little is known about its close relative SHOX2. We report the restricted expression of Shox2 in the anterior domain of the secondary palate in mice and humans. Shox2-/- mice develop an incomplete cleft that is confined to the anterior region of the palate, an extremely rare type of clefting in humans. The Shox2-/- palatal shelves initiate, grow and elevate normally, but the anterior region fails to contact and fuse at the midline, owing to altered cell proliferation and apoptosis, leading to incomplete clefting within the presumptive hard palate. Accompanied with these cellular alterations is an ectopic expression of Fgf10 and Fgfr2c in the anterior palatal mesenchyme of the mutants. Tissue recombination and bead implantation experiments revealed that signals from the anterior palatal epithelium are responsible for the restricted mesenchymal Shox2 expression. BMP activity is necessary but not sufficient for the induction of palatal Shox2 expression. Our results demonstrate an intrinsic requirement for Shox2 in palatogenesis, and support the idea that palatogenesis is differentially regulated along the anteroposterior axis. Furthermore, our results demonstrate that fusion of the posterior palate can occur independently of fusion in the anterior palate.
Ling Yu; Shuping Gu; Sylvia Alappat; Yiqiang Song; Mingquan Yan; Xiaoyun Zhang; Guozhong Zhang; Yiping Jiang; Zunyi Zhang; Yanding Zhang; YiPing Chen
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.     Date:  2005-09-01
Journal Detail:
Title:  Development (Cambridge, England)     Volume:  132     ISSN:  0950-1991     ISO Abbreviation:  Development     Publication Date:  2005 Oct 
Date Detail:
Created Date:  2005-09-12     Completed Date:  2005-12-16     Revised Date:  2009-11-19    
Medline Journal Info:
Nlm Unique ID:  8701744     Medline TA:  Development     Country:  England    
Other Details:
Languages:  eng     Pagination:  4397-406     Citation Subset:  IM    
Division of Developmental Biology, Department of Cell and Molecular Biology, and Center for Bioenvironmental Research, Tulane University, New Orleans, LA 70118, USA.
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MeSH Terms
Bone Morphogenetic Proteins / metabolism
Cleft Palate / embryology,  genetics,  metabolism
Fibroblast Growth Factor 10 / metabolism
Gene Expression Regulation, Developmental
Homeodomain Proteins / genetics,  metabolism*
Mesoderm / cytology,  metabolism
Mice, Knockout
Palate / embryology*,  metabolism
Receptor, Fibroblast Growth Factor, Type 2 / metabolism
Signal Transduction
Grant Support
Reg. No./Substance:
0/Bone Morphogenetic Proteins; 0/Fgf10 protein, mouse; 0/Fibroblast Growth Factor 10; 0/Homeodomain Proteins; 0/SHOX2 protein, human; 0/Shox2 protein, mouse; EC, Fibroblast Growth Factor, Type 2

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