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Gli2a protein localization reveals a role for Iguana/DZIP1 in primary ciliogenesis and a dependence of Hedgehog signal transduction on primary cilia in the zebrafish

Hyejeong Rosemary Kim1, Joanna Richardson12, Freek van Eeden1 and Philip W Ingham13*

Author Affiliations

1 MRC Centre for Developmental & Biomedical Genetics, University of Sheffield, Sheffield S10 2TN, UK

2 Current Address: MRC Centre for Developmental Neurobiology, New Hunt's House, Guy's Campus, London SE1 1UL, UK

3 Current Address: Institute of Molecular and Cell Biology, Proteos, 61 Biopolis Drive, Singapore 138673

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BMC Biology 2010, 8:65  doi:10.1186/1741-7007-8-65

Published: 19 April 2010



In mammalian cells, the integrity of the primary cilium is critical for proper regulation of the Hedgehog (Hh) signal transduction pathway. Whether or not this dependence on the primary cilium is a universal feature of vertebrate Hedgehog signalling has remained contentious due, in part, to the apparent divergence of the intracellular transduction pathway between mammals and teleost fish.


Here, using a functional Gli2-GFP fusion protein, we show that, as in mammals, the Gli2 transcription factor localizes to the primary cilia of cells in the zebrafish embryo and that this localization is modulated by the activity of the Hh pathway. Moreover, we show that the Igu/DZIP1protein, previously implicated in the modulation of Gli activity in zebrafish, also localizes to the primary cilium and is required for its proper formation.


Our findings demonstrate a conserved role of the primary cilium in mediating Hedgehog signalling activity across the vertebrate phylum and validate the use of the zebrafish as a representative model for the in vivo analysis of vertebrate Hedgehog signalling.