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- W2111519064 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." @default.
- W2111519064 created "2016-06-24" @default.
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- W2111519064 date "2009-10-01" @default.
- W2111519064 modified "2023-10-18" @default.
- W2111519064 title "Craniofacial skeletal defects of adult zebrafish<i>Glypican 4 (knypek)</i>mutants" @default.
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- W2111519064 doi "https://doi.org/10.1002/dvdy.22086" @default.
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