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Zebrafish ift57, ift88, and ift172 intraflagellar transport mutants disrupt cilia but do not affect hedgehog signaling.

Cilia formation requires intraflagellar transport (IFT) proteins. Recent studies indicate that mammalian Hedgehog (Hh) signaling requires cilia. It is unclear, however, if the requirement for cilia and IFT proteins in Hh signaling represents a general rule for all vertebrates. Here we examine zebrafish ift57, ift88, and ift172 mutants and morphants for defects in Hh signaling. Although ift57 and ift88 mutants and morphants contained residual maternal protein, the cilia were disrupted. In contrast to previous genetic studies in mouse, mutations in zebrafish IFT genes did not affect the expression of Hh target genes in the neural tube and forebrain and had no quantitative effect on Hh target gene expression. Zebrafish IFT mutants also exhibited no dramatic changes in the craniofacial skeleton, somite formation, or motor neuron patterning. Thus, our data indicate the requirement for cilia in the Hh signal transduction pathway may not represent a universal mechanism in vertebrates.

Pubmed ID: 19517571


  • Lunt SC
  • Haynes T
  • Perkins BD


Developmental dynamics : an official publication of the American Association of Anatomists

Publication Data

July 24, 2009

Associated Grants

  • Agency: NEI NIH HHS, Id: EY017037
  • Agency: NEI NIH HHS, Id: R01 EY017037
  • Agency: NEI NIH HHS, Id: R01 EY017037-04

Mesh Terms

  • Adaptor Proteins, Signal Transducing
  • Animals
  • Animals, Genetically Modified
  • Carrier Proteins
  • Cilia
  • Embryo, Nonmammalian
  • Gene Expression Regulation, Developmental
  • Gene Knockdown Techniques
  • Hedgehog Proteins
  • Mutant Proteins
  • Mutation
  • Signal Transduction
  • Veratrum Alkaloids
  • Zebrafish
  • Zebrafish Proteins