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Development 136 (18): 3089-3098

Dampened Hedgehog signaling but normal Wnt signaling in zebrafish without cilia

Peng Huang*, and Alexander F. Schier*

Department of Molecular and Cellular Biology, Center for Brain Science, Harvard Stem Cell Institute, Broad Institute, Harvard University, 16 Divinity Avenue, Cambridge, MA 02138, USA.

* Authors for correspondence (huang{at}; schier{at}

Accepted for publication 22 July 2009.

Abstract: 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.

Key Words: Cilia • Hedgehog signaling • Wnt signaling • Gli • Spinal cord • Somite • Zebrafish

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