ZFIN ID: ZDB-PUB-160601-3
Early Craniofacial Defects in Zebrafish That Have Reduced Function of a Wnt-Interacting Extracellular Matrix Protein, Tinagl1
Neiswender, H., Navarre, S., Kozlowski, D.J., LeMosy, E.K.
Date: 2017
Source: The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association   54(4): 381-390 (Journal)
Registered Authors: Kozlowski, David J., LeMosy, Ellen K., Navarre, Sammy
Keywords: Tinagl1, Wnt3a, dlx2a, neural crest cells, zebrafish
MeSH Terms:
  • Animals
  • Branchial Region/abnormalities*
  • Branchial Region/metabolism*
  • Cartilage/abnormalities*
  • Cartilage/metabolism*
  • Cell Movement
  • Cell Proliferation
  • Craniofacial Abnormalities/genetics
  • Craniofacial Abnormalities/metabolism*
  • Embryo, Nonmammalian/metabolism
  • Extracellular Matrix Proteins/chemistry
  • Extracellular Matrix Proteins/genetics
  • Extracellular Matrix Proteins/metabolism*
  • Gene Expression Regulation, Developmental
  • Gene Knockdown Techniques
  • In Situ Hybridization
  • Lipocalins/chemistry
  • Lipocalins/genetics
  • Lipocalins/metabolism*
  • Polymerase Chain Reaction
  • Wnt3A Protein/chemistry
  • Wnt3A Protein/genetics
  • Wnt3A Protein/metabolism*
  • Zebrafish/embryology*
  • Zebrafish/genetics
  • Zebrafish Proteins/chemistry
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
PubMed: 27243669 Full text @ Cleft Palate Craniofac. J.
  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.
  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.
  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.