Journal
DEVELOPMENT
Volume 134, Issue 14, Pages 2569-2577Publisher
COMPANY OF BIOLOGISTS LTD
DOI: 10.1242/dev.003715
Keywords
basal body; left-right asymmetry; limb development; mouse mutant; neural tube
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Funding
- NCI NIH HHS [R01 CA111673] Funding Source: Medline
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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.
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