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Early Craniofacial Defects in Zebrafish That Have Reduced Function of a Wnt-Interacting Extracellular Matrix Protein, Tinagl1.
- Source :
-
The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association [Cleft Palate Craniofac J] 2017 Jul; Vol. 54 (4), pp. 381-390. Date of Electronic Publication: 2016 May 31. - Publication Year :
- 2017
-
Abstract
- Objective: Tinagl1 has a weak genetic association with craniosynostosis, but its functions in cartilage and bone development are unknown. Knockdown of Tinagl1 in zebrafish embryos allowed an initial characterization of its potential effects on craniofacial cartilage development and a test of whether these effects could involve Wnt signaling.<br />Results: Tinagl1 knockdown resulted in dose-dependent reductions and defects in ventral pharyngeal arch cartilages as well as the ethmoid plate, a zebrafish correlate to the palate. These defects could be correlated to reduced numbers of cranial neural crest cells in the pharyngeal arches and could be reproduced with comanipulation of Tinagl1 and Wnt3a by morpholino-based knockdown.<br />Conclusions: These results suggest that Tinagl1 is required early in the proliferation or migration of cranial neural crest cells and that its effects are mediated via Wnt3a signaling. Because Wnt3a is among the Wnts that contribute to nonsyndromic cleft lip and cleft palate in mouse and man, further investigation of Tinagl1 may help to elucidate mechanisms underlying these disorders.
- Subjects :
- Animals
Cell Movement
Cell Proliferation
Craniofacial Abnormalities genetics
Embryo, Nonmammalian metabolism
Extracellular Matrix Proteins chemistry
Extracellular Matrix Proteins genetics
Gene Expression Regulation, Developmental
Gene Knockdown Techniques
In Situ Hybridization
Lipocalins chemistry
Lipocalins genetics
Polymerase Chain Reaction
Wnt3A Protein chemistry
Wnt3A Protein genetics
Zebrafish genetics
Zebrafish Proteins chemistry
Zebrafish Proteins genetics
Branchial Region abnormalities
Branchial Region metabolism
Cartilage abnormalities
Cartilage metabolism
Craniofacial Abnormalities metabolism
Extracellular Matrix Proteins metabolism
Lipocalins metabolism
Wnt3A Protein metabolism
Zebrafish embryology
Zebrafish Proteins metabolism
Subjects
Details
- Language :
- English
- ISSN :
- 1545-1569
- Volume :
- 54
- Issue :
- 4
- Database :
- MEDLINE
- Journal :
- The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
- Publication Type :
- Academic Journal
- Accession number :
- 27243669
- Full Text :
- https://doi.org/10.1597/15-283