title: Functional genomics of the cilium, a sensory organelle.
abstract: Cilia and flagella play important roles in many physiological processes, including cell and fluid movement, sensory perception, and development. The biogenesis and maintenance of cilia depend on intraflagellar transport (IFT), a motility process that operates bidirectionally along the ciliary axoneme. Disruption in IFT and cilia function causes several human disorders, including polycystic kidneys, retinal dystrophy, neurosensory impairment, and Bardet-Biedl syndrome (BBS). 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 ciliogenic transcription factor, DAF-19. 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.
authors.affiliation: Department of Molecular Biology and Biochemistry, Simon Fraser University, 8888 University Drive, Burnaby, British Columbia V5A 1S6, Canada.
authors.forename: Oliver E
authors.initials: OE
authors.lastname: Blacque
authors.forename: Elliot A
authors.initials: EA
authors.lastname: Perens
authors.forename: Keith A
authors.initials: KA
authors.lastname: Boroevich
authors.forename: Peter N
authors.initials: PN
authors.lastname: Inglis
authors.forename: Chunmei
authors.initials: C
authors.lastname: Li
authors.forename: Adam
authors.initials: A
authors.lastname: Warner
authors.forename: Jaswinder
authors.initials: J
authors.lastname: Khattra
authors.forename: Rob A
authors.initials: RA
authors.lastname: Holt
authors.forename: Guangshuo
authors.initials: G
authors.lastname: Ou
authors.forename: Allan K
authors.initials: AK
authors.lastname: Mah
authors.forename: Sheldon J
authors.initials: SJ
authors.lastname: McKay
authors.forename: Peter
authors.initials: P
authors.lastname: Huang
authors.forename: Peter
authors.initials: P
authors.lastname: Swoboda
authors.forename: Steve J M
authors.initials: SJ
authors.lastname: Jones
authors.forename: Marco A
authors.initials: MA
authors.lastname: Marra
authors.forename: David L
authors.initials: DL
authors.lastname: Baillie
authors.forename: Donald G
authors.initials: DG
authors.lastname: Moerman
authors.forename: Shai
authors.initials: S
authors.lastname: Shaham
authors.forename: Michel R
authors.initials: MR
authors.lastname: Leroux
mesh_terms: D000818:Animals; D001483:Base Sequence; D017173:Caenorhabditis elegans / Q000235:genetics*; D029742:Caenorhabditis elegans Proteins / Q000378:metabolism; D002923:Cilia / Q000235:genetics* / Q000378:metabolism; D019295:Computational Biology; D020869:Gene Expression Profiling; D023281:Genomics / Q000379:methods; D049452:Green Fluorescent Proteins; D009154:Mutation / Q000235:genetics; D009474:Neurons / Q000378:metabolism*; D021381:Protein Transport / Q000502:physiology; D017422:Sequence Analysis, DNA; D014157:Transcription Factors / Q000378:metabolism
chemical_list: D029742:Caenorhabditis elegans Proteins; C410596:DAF-19 protein, C elegans; D014157:Transcription Factors; D049452:Green Fluorescent Proteins