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Dev Dyn. 2009 Oct;238(10):2550-63. doi: 10.1002/dvdy.22086.

Craniofacial skeletal defects of adult zebrafish Glypican 4 (knypek) mutants.

Author information

1
Department of Biological Sciences, DePaul University, Chicago, Illinois 60614, USA.

Abstract

The heparan sulfate proteoglycan Glypican 4 (Gpc4) is part of the Wnt/planar cell polarity pathway, which is required for convergence and extension during zebrafish gastrulation. To observe Glypican 4-deficient phenotypes at later stages, we rescued gpc4(-/-) (knypek) homozygotes and raised them for more than one year. Adult mutants showed diverse cranial malformations of both dermal and endochondral bones, ranging from shortening of the rostral-most skull to loss of the symplectic. Additionally, the adult palatoquadrate cartilage was disorganized, with abnormal chondrocyte orientation. To understand how the palatoquadrate cartilage normally develops, we examined a juvenile series of wild type and mutant specimens. This identified two novel domains of elongated chondrocytes in the larval palatoquadrate, which normally form prior to endochondral ossification. In contrast, gpc4(-/-) larvae never form these domains, suggesting a failure of chondrocyte orientation, though not differentiation. Our findings implicate Gpc4 in the regulation of zebrafish cartilage and bone morphogenesis.

PMID:
19777561
PMCID:
PMC3011836
DOI:
10.1002/dvdy.22086
[Indexed for MEDLINE]
Free PMC Article

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