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First published online 6 June 2007
doi: 10.1242/dev.003715

Development 134, 2569-2577 (2007)
Published by The Company of Biologists 2007
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Ftm is a novel basal body protein of cilia involved in Shh signalling

Jeanette Vierkotten1, Renate Dildrop1, Thomas Peters1,*, Baolin Wang2 and Ulrich Rüther1,{dagger}

1 Institut für Entwicklungs- und Molekularbiologie der Tiere (EMT), Heinrich-Heine-Universität, 40225 Düsseldorf, Germany.
2 Department of Genetic Medicine, Weill Medical College of Cornell University, New York, NY 10021, USA.

{dagger} Author for correspondence (e-mail: ruether{at}uni-duesseldorf.de)

Accepted 14 May 2007

In this study we show in mice that Ftm (Rpgrip1l) is located at the ciliary basal body. Our data reveal that Ftm is necessary for developmental processes such as the establishment of left-right asymmetry and patterning of the neural tube and the limbs. The loss of Ftm affects the ratio of Gli3 activator to Gli3 repressor, suggesting an involvement of Ftm in Shh signalling. As Ftm is not essential for cilia assembly but for full Shh response, Ftm can be considered as a novel component for cilium-related Hh signalling. Furthermore, the absence of Ftm in arthropods underlines the divergence between vertebrate and Drosophila Hh pathways.

Key words: Basal body, Left-right asymmetry, Limb development, Mouse mutant, Neural tube


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