Zebrafish ift57, ift88, and ift172 intraflagellar transport mutants disrupt cilia but do not affect hedgehog signaling
Open Access
- 10 June 2009
- journal article
- research article
- Published by Wiley in Developmental Dynamics
- Vol. 238 (7), 1744-1759
- https://doi.org/10.1002/dvdy.21999
Abstract
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. Developmental Dynamics 238:1744–1759, 2009.Keywords
This publication has 90 references indexed in Scilit:
- Convergent extension movements and ciliary function are mediated by ofd1 , a zebrafish orthologue of the human oral-facial-digital type 1 syndrome geneHuman Molecular Genetics, 2008
- The intraflagellar transport protein IFT57 is required for cilia maintenance and regulates IFT-particle–kinesin-II dissociation in vertebrate photoreceptorsJournal of Cell Science, 2008
- Inhibition of neural crest migration underlies craniofacial dysmorphology and Hirschsprung's disease in Bardet–Biedl syndromeProceedings of the National Academy of Sciences of the United States of America, 2008
- The Oak Ridge Polycystic Kidney mouse: Modeling ciliopathies of mice and menDevelopmental Dynamics, 2008
- Identification of a Novel BBS Gene (BBS12) Highlights the Major Role of a Vertebrate-Specific Branch of Chaperonin-Related Proteins in Bardet-Biedl SyndromeAmerican Journal of Human Genetics, 2007
- Vertebrate Smoothened functions at the primary ciliumNature, 2005
- Hedgehog signalling in the mouse requires intraflagellar transport proteinsNature, 2003
- Intraflagellar transportNature Reviews Molecular Cell Biology, 2002
- Stages of embryonic development of the zebrafishDevelopmental Dynamics, 1995
- A motility in the eukaryotic flagellum unrelated to flagellar beating.Proceedings of the National Academy of Sciences of the United States of America, 1993