Journal article
Prickle1 stunts limb growth through alteration of cell polarity and gene expression
Developmental dynamics, Vol.242(11), pp.1293-1306
11/2013
DOI: 10.1002/dvdy.24025
PMCID: PMC3985166
PMID: 23913870
Abstract
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.
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.
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.
Details
- Title: Subtitle
- Prickle1 stunts limb growth through alteration of cell polarity and gene expression
- Creators
- Tian Yang - Department of Biology, University of Iowa, Iowa City, IowaAlexander G BassukBernd Fritzsch
- Resource Type
- Journal article
- Publication Details
- Developmental dynamics, Vol.242(11), pp.1293-1306
- DOI
- 10.1002/dvdy.24025
- PMID
- 23913870
- PMCID
- PMC3985166
- NLM abbreviation
- Dev Dyn
- ISSN
- 1058-8388
- eISSN
- 1097-0177
- Publisher
- United States
- Grant note
- 1R01 NS064159-01A1 / NINDS NIH HHS R01 NS064159 / NINDS NIH HHS P30 DC 010362 / NIDCD NIH HHS R01 DC005590 / NIDCD NIH HHS P30 DC010362 / NIDCD NIH HHS
- Language
- English
- Date published
- 11/2013
- Academic Unit
- Neurology; Stead Family Department of Pediatrics; Iowa Neuroscience Institute; Biology; Craniofacial Anomalies Research Center; Neurology (Pediatrics)
- Record Identifier
- 9984020747802771
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