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| Content Provider | PubMed Central |
|---|---|
| Author | Wilson, Christopher W. Nguyen, Catherine T. Chen, Miao-hsueh Yang, Jehn-hsiahn Gacayan, Rhodora Huang, Jie Chen, Jau-nian Chuang, Pao-tien |
| Abstract | Hedgehog (Hh) signaling is essential for multiple aspects of embryogenesis1, 2. In Drosophila, Hh transduction is mediated by a cytoplasmic signaling complex3-5 that includes the putative serinethreonine kinase Fused (Fu) and the kinesin Costal 2 (Cos2), yet Fu does not play a conserved role in Hh signaling in mammals6, 7. Mouse Fu mutants are viable and appear to respond normally to Hh signaling. Here we show that mouse Fu is essential for construction of the central pair (CP) apparatus of motile, 9+2 cilia and offers a novel model of human primary ciliary dyskinesia. We found that mouse Fu physically interacts with Kif27, a mammalian Cos2 ortholog8, and linked Fu to known structural components of the CP apparatus, providing evidence for the first regulatory component involved in CP construction. We also demonstrated that zebrafish Fu is required both for Hh signaling and cilia biogenesis in Kupffer’s vesicle. Mouse Fu rescued both Hh-dependent and independent defects in zebrafish. Our results delineate a novel pathway for CP apparatus assembly, identify common regulators of Hh signaling and motile ciliogenesis, and add insight into evolution of the Hh cascade. |
| Related Links | http://dx.doi.org/10.1038/nature07883 |
| Ending Page | 102 |
| Page Count | 5 |
| Starting Page | 98 |
| File Format | |
| ISSN | 00280836 |
| e-ISSN | 14764687 |
| Journal | Nature |
| Issue Number | 7243 |
| Volume Number | 459 |
| Language | English |
| Publisher Date | 2009-05-07 |
| Access Restriction | Open |
| Subject Keyword | General Research in Higher Education |
| Content Type | Text |
| Resource Type | Article |
| Subject | Multidisciplinary |
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