ZFIN ID: ZDB-PUB-160315-9
Pdgfra and Pdgfrb genetically interact during craniofacial development
McCarthy, N., Liu, J.S., Richarte, A.M., Eskiocak, B., Lovely, C.B., Tallquist, M.D., Eberhart, J.K.
Date: 2016
Source: Developmental dynamics : an official publication of the American Association of Anatomists   245(6): 641-52 (Journal)
Registered Authors: Eberhart, Johann, McCarthy, Neil
Keywords: cranial neural crest, palatogenesis, pdgfra, pdgfrb
MeSH Terms:
  • Animals
  • Cleft Palate/embryology
  • Cleft Palate/genetics
  • Cleft Palate/metabolism
  • Female
  • Gene Expression Regulation, Developmental/genetics
  • Gene Expression Regulation, Developmental/physiology
  • Immunohistochemistry
  • In Situ Hybridization
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Neural Crest/embryology
  • Neural Crest/metabolism
  • Receptor, Platelet-Derived Growth Factor alpha/genetics
  • Receptor, Platelet-Derived Growth Factor alpha/metabolism*
  • Receptor, Platelet-Derived Growth Factor beta/genetics
  • Receptor, Platelet-Derived Growth Factor beta/metabolism*
  • Signal Transduction/genetics
  • Signal Transduction/physiology
  • Zebrafish/embryology
  • Zebrafish/genetics
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
PubMed: 26971580 Full text @ Dev. Dyn.
One of the most prevalent congenital birth defects is cleft palate. The palatal skeleton is derived from the cranial neural crest and platelet-derived growth factors (Pdgf) are critical in palatogenesis. Of the two Pdgf receptors, pdgfra is required for neural crest migration and palatogenesis. However, the role pdgfrb plays in the neural crest, or whether pdgfra and pdgfrb interact during palatogenesis is unclear.
We find that pdgfrb is dispensable for craniofacial development in zebrafish. However, the palatal defect in pdgfra;pdgfrb double mutants is significantly more severe than in pdgfra single mutants. Data in mouse suggest this interaction is conserved and that neural crest requires both genes. In zebrafish, pdgfra and pdgfrb are both expressed by neural crest within the pharyngeal arches and pharmacological analyses demonstrate Pdgf signaling is required at these times. While neither proliferation nor cell death appears affected, time-lapsed confocal analysis of pdgfra;pdgfrb mutants shows a failure of proper neural crest condensation during palatogenesis.
We provide data showing that pdgfra and pdgfrb interact during palatogenesis in both zebrafish and mouse. In zebrafish, this interaction affects proper condensation of maxillary neural crest cells, revealing a previously unknown interaction between Pdgfra and Pdgfrb during palate formation. This article is protected by copyright. All rights reserved.