A role of glypican4 and wnt5b in chondrocyte stacking underlying craniofacial cartilage morphogenesis
- Sisson, B.E., Dale, R.M., Mui, S.R., Topczewska, J.M., Topczewski, J.
- Mechanisms of Development 138 Pt 3: 279-90 (Journal)
- Registered Authors
- Dale, Rodney M., Mui, Stephanie, Sisson, Barbara E., Topczewska, Jolanta, Topczewski, Jacek
- Growth plate, Wnt/PCP pathway, knypek, pipe tail
- MeSH Terms
- Animals, Genetically Modified
- Branchial Region/embryology
- Branchial Region/metabolism
- Cell Count
- Cell Movement/genetics
- Cell Size
- Gene Expression Regulation, Developmental
- Neural Crest/embryology
- Neural Crest/metabolism
- Wnt Proteins/deficiency
- Wnt Proteins/genetics*
- Wnt Signaling Pathway/genetics
- Wnt-5a Protein
- Zebrafish Proteins/deficiency
- Zebrafish Proteins/genetics*
- 26459057 Full text @ Mech. Dev.
Sisson, B.E., Dale, R.M., Mui, S.R., Topczewska, J.M., Topczewski, J. (2015) A role of glypican4 and wnt5b in chondrocyte stacking underlying craniofacial cartilage morphogenesis. Mechanisms of Development. 138 Pt 3:279-90.
The Wnt/Planar Cell Polarity (PCP) pathway controls cell morphology and behavior during animal development. Several zebrafish mutants were identified as having perturbed Wnt/PCP signaling. Many of these mutants have defects in craniofacial formation. To better understand the role that Wnt/PCP plays in craniofacial development we set out to identify which of the mutants, known to be associated with the Wnt/PCP pathway, perturb head cartilage formation by disrupting chondrocyte morphology. Here we demonstrate that while vang-like 2 (vangl2), wnt11 and scribbled (scrib) mutants have severe craniofacial morphogenesis defects they do not display the chondrocyte stacking and intercalation problems seen in glypican 4 (gpc4) and wnt5b mutants. The function of Gpc4 or Wnt5b appears to be important for chondrocyte organization, as the neural crest in both mutants is specified, undergoes migration, and differentiates into the same number of cells to compose the craniofacial cartilage elements. We demonstrate that Gpc4 activity is required cell autonomously in the chondrocytes and that the phenotype of single heterozygous mutants is slightly enhanced in embryos double heterozygous for wnt5b and gpc4. This data suggests a novel mechanism for Wnt5b and Gpc4 regulation of chondrocyte behavior that is independent of the core Wnt/PCP molecules and differs from their collaborative action of controlling cell movements during gastrulation.
Genes / Markers
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Engineered Foreign Genes
Errata and Notes