PUBLICATION
Craniofacial skeletal defects of adult zebrafish Glypican 4 (knypek) mutants
- Authors
- LeClair, E.E., Mui, S.R., Huang, A., Topczewska, J.M., and Topczewski, J.
- ID
- ZDB-PUB-090929-7
- Date
- 2009
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists 238(10): 2550-2563 (Journal)
- Registered Authors
- LeClair, Elizabeth E., Mui, Stephanie, Topczewska, Jolanta, Topczewski, Jacek
- Keywords
- zebrafish, glypican, knypek, craniofacial, skull, jaw, bone, cartilage, morphometrics
- MeSH Terms
-
- Animals
- Animals, Genetically Modified
- Body Patterning*
- Cartilage*/abnormalities
- Cartilage*/anatomy & histology
- Cartilage*/growth & development
- Facial Bones*/abnormalities
- Facial Bones*/anatomy & histology
- Facial Bones*/growth & development
- Gene Knockdown Techniques
- Glypicans/genetics
- Glypicans/metabolism*
- Humans
- Phenotype
- Skull*/abnormalities
- Skull*/anatomy & histology
- Skull*/growth & development
- Zebrafish*/abnormalities
- Zebrafish*/anatomy & histology
- Zebrafish*/growth & development
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism*
- PubMed
- 19777561 Full text @ Dev. Dyn.
Citation
LeClair, E.E., Mui, S.R., Huang, A., Topczewska, J.M., and Topczewski, J. (2009) Craniofacial skeletal defects of adult zebrafish Glypican 4 (knypek) mutants. Developmental Dynamics : an official publication of the American Association of Anatomists. 238(10):2550-2563.
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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping