Document Detail

Expression of Wnt9b and activation of canonical Wnt signaling during midfacial morphogenesis in mice.
MedLine Citation:
PMID:  16496313     Owner:  NLM     Status:  MEDLINE    
Cleft lip with or without cleft palate (CLP) is the most common craniofacial birth defect in humans. Recently, mutations in the WNT3 and Wnt9b genes, encoding two members of the Wnt family of signaling molecules, were found associated with CLP in human and mice, respectively. To investigate whether Wnt3 and Wnt9b directly regulate facial development, we analyzed their developmental expression patterns and found that both Wnt3 and Wnt9b are expressed in the facial ectoderm at critical stages of midfacial morphogenesis during mouse embryogenesis. Whereas Wnt3 mRNA is mainly expressed in the maxillary and medial nasal ectoderm, Wnt9b mRNA is expressed in maxillary, medial nasal, and lateral nasal ectoderm. During lip fusion, Wnt9b, but not Wnt3, is expressed in the epithelial seam between the fusing medial and lateral nasal processes. Furthermore, we found that expression of TOPGAL, a transgenic reporter of activation of canonical Wnt signaling pathway, is specifically activated in the distal regions of the medial nasal, lateral nasal, and maxillary processes prior to lip fusion. During lip fusion, the epithelial seam between the medial and lateral nasal processes as well as the facial mesenchyme directly beneath the fusing epithelia strongly expresses TOPGAL. These data, together with the CLP lip phenotype in WNT3-/- humans and Wnt9b-/- mutant mice, indicate that Wnt3 and Wnt9b signal through the canonical Wnt signaling pathway to regulate midfacial development and lip fusion.
Yu Lan; Rosemary C Ryan; Zunyi Zhang; Steven A Bullard; Jeffrey O Bush; Kathleen M Maltby; Andrew C Lidral; Rulang Jiang
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural    
Journal Detail:
Title:  Developmental dynamics : an official publication of the American Association of Anatomists     Volume:  235     ISSN:  1058-8388     ISO Abbreviation:  Dev. Dyn.     Publication Date:  2006 May 
Date Detail:
Created Date:  2006-04-27     Completed Date:  2006-12-11     Revised Date:  2014-09-14    
Medline Journal Info:
Nlm Unique ID:  9201927     Medline TA:  Dev Dyn     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1448-54     Citation Subset:  IM    
Copyright Information:
(c) 2006 Wiley-Liss, Inc.
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MeSH Terms
Cleft Lip / embryology
Cleft Palate / embryology
Glycoproteins / biosynthesis,  genetics*,  physiology
Lip / embryology*
Signal Transduction / physiology*
Wnt Proteins / biosynthesis,  genetics*,  physiology
Wnt3 Protein
Grant Support
DE015207/DE/NIDCR NIH HHS; K02 DE015291/DE/NIDCR NIH HHS; K02 DE015291-01/DE/NIDCR NIH HHS; K02 DE015291-02/DE/NIDCR NIH HHS; K02 DE015291-03/DE/NIDCR NIH HHS; K02 DE015291-04/DE/NIDCR NIH HHS; K02 DE015291-05/DE/NIDCR NIH HHS; KO2DE015291/DE/NIDCR NIH HHS; P50 DE016215/DE/NIDCR NIH HHS; P50DE016215/DE/NIDCR NIH HHS; R01 DE014667/DE/NIDCR NIH HHS; R01 DE014667-01/DE/NIDCR NIH HHS; R01 DE014667-02/DE/NIDCR NIH HHS; R01 DE014667-03/DE/NIDCR NIH HHS; R01 DE014667-04/DE/NIDCR NIH HHS; R01 DE014667-05/DE/NIDCR NIH HHS; R01 DE014667-06/DE/NIDCR NIH HHS; R01 DE014667-07/DE/NIDCR NIH HHS; R01 DE014667-08/DE/NIDCR NIH HHS; R01 DE015207/DE/NIDCR NIH HHS; R01 DE015207-05A1/DE/NIDCR NIH HHS
Reg. No./Substance:
0/Glycoproteins; 0/WNT3 protein, human; 0/Wnt Proteins; 0/Wnt3 Protein; 0/Wnt3 protein, mouse; 0/Wnt9b protein, mouse

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