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

Elizabeth E. LeClair*, Stephanie R. Mui, Angela Huang, Jolanta M. Topczewska, Jacek Topczewski

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

42 Scopus citations

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.

Original languageEnglish (US)
Pages (from-to)2550-2563
Number of pages14
JournalDevelopmental dynamics : an official publication of the American Association of Anatomists
Volume238
Issue number10
DOIs
StatePublished - Oct 2009

ASJC Scopus subject areas

  • Developmental Biology

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