Document Detail


Prickle1 stunts limb growth through alteration of cell polarity and gene expression.
MedLine Citation:
PMID:  23913870     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: Wnt/PCP signaling plays a critical role in multiple developmental processes, including limb development. Wnt5a, a ligand of the PCP pathway, signals through the Ror2/Vangl2 or the Vangl2/Ryk complex to regulate limb development along the proximal-distal axis in mice. Based on the interaction between Van Gogh and Prickle in Drosophila, we hypothesized the vertebrate Prickle1 has a similar function as Vangl2 in limb development.
RESULTS: We show Prickle1 is expressed in the skeletal condensates that will differentiate into chondrocytes and later form bones. Disrupted Prickle1 function in Prickle1(C251X/C251X) mouse mutants alters expression of genes such as Bmp4, Fgf8, Vangl2, and Wnt5a. These expression changes correlate with shorter and wider bones in the limbs and loss of one phalangeal segment in digits 2-5 of Prickle1C251X mutants. These growth defects along the proximal-distal axis are also associated with increased cell death in the growing digit tip, reduced cell death in the interdigital membrane, and disrupted chondrocyte polarity.
CONCLUSIONS: We suggest Prickle1 is part of the Wnt5a/PCP signaling, regulating cell polarity and affecting expression of multiple factors to stunt limb growth through altered patterns of gene expression, including the PCP genes Wnt5a and Vangl2.
Authors:
Tian Yang; Alexander G Bassuk; Bernd Fritzsch
Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural     Date:  2013-09-06
Journal Detail:
Title:  Developmental dynamics : an official publication of the American Association of Anatomists     Volume:  242     ISSN:  1097-0177     ISO Abbreviation:  Dev. Dyn.     Publication Date:  2013 Nov 
Date Detail:
Created Date:  2013-10-24     Completed Date:  2014-07-17     Revised Date:  2014-11-04    
Medline Journal Info:
Nlm Unique ID:  9201927     Medline TA:  Dev Dyn     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1293-306     Citation Subset:  IM    
Copyright Information:
Copyright © 2013 Wiley Periodicals, Inc.
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MeSH Terms
Descriptor/Qualifier:
Adaptor Proteins, Signal Transducing / genetics,  metabolism*
Animals
Bone Morphogenetic Protein 4 / genetics,  metabolism
Cell Polarity / genetics,  physiology*
Chondrocytes / cytology,  metabolism
Extremities / embryology*
Fibroblast Growth Factor 8 / genetics,  metabolism
LIM Domain Proteins / genetics,  metabolism*
Mice
Nerve Tissue Proteins / genetics,  metabolism
Signal Transduction / genetics,  physiology
Wnt Proteins / genetics,  metabolism
Grant Support
ID/Acronym/Agency:
1R01 NS064159-01A1/NS/NINDS NIH HHS; P30 DC 010362/DC/NIDCD NIH HHS; P30 DC010362/DC/NIDCD NIH HHS; R01 DC005590/DC/NIDCD NIH HHS; R01 DC005590/DC/NIDCD NIH HHS
Chemical
Reg. No./Substance:
0/Adaptor Proteins, Signal Transducing; 0/Bmp4 protein, mouse; 0/Bone Morphogenetic Protein 4; 0/Fgf8 protein, mouse; 0/LIM Domain Proteins; 0/Ltap protein, mouse; 0/Nerve Tissue Proteins; 0/Prickle1 protein, mouse; 0/Wnt Proteins; 0/Wnt5a protein, mouse; 148997-75-5/Fibroblast Growth Factor 8
Comments/Corrections

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