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Development DOI:10.1242/dev.041343

Dampened Hedgehog signaling but normal Wnt signaling in zebrafish without cilia.

Publication TypeJournal Article
Year of Publication2009
AuthorsHuang, P, Schier, AF
Date Published2009 Sep
KeywordsAnimals, Animals, Genetically Modified, Body Patterning, Cilia, Epistasis, Genetic, Hedgehog Proteins, In Situ Hybridization, Oligonucleotides, Antisense, Oncogene Proteins, Signal Transduction, Trans-Activators, Wnt Proteins, Zebrafish, Zebrafish Proteins, Zinc Finger Protein GLI1

Cilia have been implicated in Hedgehog (Hh) and Wnt signaling in mouse but not in Drosophila. To determine whether the role of cilia is conserved in zebrafish, we generated maternal-zygotic (MZ) oval (ovl; ift88) mutants that lack all cilia. MZovl mutants display normal canonical and non-canonical Wnt signaling but show defects in Hh signaling. As in mouse, zebrafish cilia are required to mediate the activities of Hh, Ptc, Smo and PKA. However, in contrast to mouse Ift88 mutants, which show a dramatic reduction in Hh signaling, zebrafish MZovl mutants display dampened, but expanded, Hh pathway activity. This activity is largely due to gli1, the expression of which is fully dependent on Hh signaling in mouse but not in zebrafish. These results reveal a conserved requirement for cilia in transducing the activity of upstream regulators of Hh signaling but distinct phenotypic effects due to differential regulation and differing roles of transcriptional mediators.


Alternate JournalDevelopment
PubMed ID19700616
PubMed Central IDPMC2730366
Grant ListR01 DE016779 / DE / NIDCR NIH HHS / United States
R01 GM056211 / GM / NIGMS NIH HHS / United States