Examination of a palatogenic gene program in zebrafish

Dev Dyn. 2011 Sep;240(9):2204-20. doi: 10.1002/dvdy.22713.

Abstract

Human palatal clefting is debilitating and difficult to rectify surgically. Animal models enhance our understanding of palatogenesis and are essential in strategies designed to ameliorate palatal malformations in humans. Recent studies have shown that the zebrafish palate, or anterior neurocranium, is under similar genetic control to the amniote palatal skeleton. We extensively analyzed palatogenesis in zebrafish to determine the similarity of gene expression and function across vertebrates. By 36 hours postfertilization (hpf) palatogenic cranial neural crest cells reside in homologous regions of the developing face compared with amniote species. Transcription factors and signaling molecules regulating mouse palatogenesis are expressed in similar domains during palatogenesis in zebrafish. Functional investigation of a subset of these genes, fgf10a, tgfb2, pax9, and smad5 revealed their necessity in zebrafish palatogenesis. Collectively, these results suggest that the gene regulatory networks regulating palatogenesis may be conserved across vertebrate species, demonstrating the utility of zebrafish as a model for palatogenesis.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Animals
  • Fibroblast Growth Factor 10 / genetics
  • Fibroblast Growth Factor 10 / metabolism
  • Gene Expression Regulation, Developmental
  • In Situ Hybridization
  • PAX9 Transcription Factor / genetics
  • PAX9 Transcription Factor / metabolism
  • Palate / embryology*
  • Palate / metabolism*
  • Signal Transduction
  • Smad5 Protein / genetics
  • Smad5 Protein / metabolism
  • Transcription Factors / genetics
  • Transcription Factors / metabolism
  • Transforming Growth Factor beta2 / genetics
  • Transforming Growth Factor beta2 / metabolism
  • Zebrafish
  • Zebrafish Proteins / genetics
  • Zebrafish Proteins / metabolism*

Substances

  • Fibroblast Growth Factor 10
  • PAX9 Transcription Factor
  • Smad5 Protein
  • Transcription Factors
  • Transforming Growth Factor beta2
  • Zebrafish Proteins
  • fgf10a protein, zebrafish
  • pax9 protein, zebrafish