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| Functional genomics of the cilium, a sensory organelle |
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| Author(s): Blacque OE, Perens EA, Boroevich KA, Inglis PN, Li CM, Warner A, Khattra J, Holt RA, Ou GS, Mah AK, McKay SJ, Huang P, Swoboda P, Jones SJM, Marra MA, Baillie DL, Moerman DG, Shaham S, Leroux MR |
| Source: CURRENT BIOLOGY Volume: 15 Issue: 10 Pages: 935-941 Published: MAY 24 2005 |
| Times Cited: 76 References: 21 |
| Abstract: Cilia and flagella play important roles in many physiological processes, including cell and fluid movement, sensory perception, and development [1]. The biogenesis and maintenance of cilia depend on intraflagellar transport (IFT), a motility process that operates bidirectionally along the ciliary axoneme [1, 2]. Disruption in IFT and cilia function causes several human disorders, including polycystic kidneys, retinal dystrophy, neurosensory impairment, and Bardet-Bledl syndrome (BBS) [3-5]. To uncover new ciliary components, including IFT proteins, we compared C. elegans ciliated neuronal and nonciliated cells through serial analysis of gene expression (SAGE) and screened for genes potentially regulated by the cillogenic transcription factor, DAF-19 [6]. Using these complementary approaches, we identified numerous candidate ciliary genes and confirmed the ciliated-cell-specific expression of 14 novel genes. One of these, C27H5.7a, encodes a ciliary protein that undergoes IFT. As with other IFT proteins, its ciliary localization and transport is disrupted by mutations in IFT and bbs genes. Furthermore, we demonstrate that the ciliary structural defect of C. elegans dyf-13(mn396) mutants is caused by a mutation in C27H5.7a. Together, our findings help define a ciliary transcriptome and suggest that DYF-13, an evolutionarily conserved protein, is a novel core IFT component required for cilia function. |
| Document Type: Article |
| Language: English |
| Reprint Address: Leroux, MR (reprint author), Simon Fraser Univ, Dept Mol Biol & Biochem, 8888 Univ Dr, Burnaby, BC V5A 1S6 Canada |
Addresses:
1. Simon Fraser Univ, Dept Mol Biol & Biochem, Burnaby, BC V5A 1S6 Canada
2. Rockefeller Univ, Lab Dev Genet, New York, NY 10021 USA
3. British Columbia Canc Agcy, Genome Sci Ctr, Vancouver, BC V5Z 4E6 Canada
4. Univ Calif Davis, Ctr Genet & Dev, Sect Mol & Cellular Biol, Davis, CA 95616 USA
5. Sodertorn Univ Coll, Karolinska Inst, Dept Biosci, Sect Nat Sci, S-14189 Huddinge, Sweden
6. Univ British Columbia, Dept Zool, Vancouver, BC V6T 1Z4 Canada |
| Publisher: CELL PRESS, 1100 MASSACHUSETTS AVE, CAMBRIDGE, MA 02138 USA |
| Subject Category: Biochemistry & Molecular Biology |
| IDS Number: 930VZ |
| ISSN: 0960-9822 |
| DOI: 10.1016/j.cub.2005.04.059 |
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