|  Help  |  About  |  Contact Us

Publication : Fused has evolved divergent roles in vertebrate Hedgehog signalling and motile ciliogenesis.

First Author  Wilson CW Year  2009
Journal  Nature Volume  459
Issue  7243 Pages  98-102
PubMed ID  19305393 Mgi Jnum  J:148415
Mgi Id  MGI:3844768 Doi  10.1038/nature07883
Citation  Wilson CW, et al. (2009) Fused has evolved divergent roles in vertebrate Hedgehog signalling and motile ciliogenesis. Nature 459(7243):98-102
abstractText  Hedgehog (Hh) signalling is essential for several aspects of embryogenesis. In Drosophila, Hh transduction is mediated by a cytoplasmic signalling complex that includes the putative serine-threonine kinase Fused (Fu) and the kinesin Costal 2 (Cos2, also known as Cos), yet Fu does not have a conserved role in Hh signalling in mammals. Mouse Fu (also known as Stk36) mutants are viable and seem to respond normally to Hh signalling. Here we show that mouse Fu is essential for construction of the central pair apparatus of motile, 9+2 cilia and offers a new model of human primary ciliary dyskinesia. We found that mouse Fu physically interacts with Kif27, a mammalian Cos2 orthologue, and linked Fu to known structural components of the central pair apparatus, providing evidence for the first regulatory component involved in central pair construction. We also demonstrated that zebrafish Fu is required both for Hh signalling and cilia biogenesis in Kupffer's vesicle. Mouse Fu rescued both Hh-dependent and -independent defects in zebrafish. Our results delineate a new pathway for central pair apparatus assembly, identify common regulators of Hh signalling and motile ciliogenesis, and provide insights into the evolution of the Hh cascade.
Quick Links:
 
Quick Links:
 

Expression

Publication --> Expression annotations

 

Other

14 Bio Entities

Trail: Publication

0 Expression