Functional genomics of the cilium, a sensory organelle.
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.
Blacque
Oliver E
OE
Department of Molecular Biology and Biochemistry, Simon Fraser University, 8888 University Drive, Burnaby, British Columbia V5A 1S6, Canada.
Perens
Elliot A
EA
Boroevich
Keith A
KA
Inglis
Peter N
PN
Li
Chunmei
C
Warner
Adam
A
Khattra
Jaswinder
J
Holt
Rob A
RA
Ou
Guangshuo
G
Mah
Allan K
AK
McKay
Sheldon J
SJ
Huang
Peter
P
Swoboda
Peter
P
Jones
Steve J M
SJ
Marra
Marco A
MA
Baillie
David L
DL
Moerman
Donald G
DG
Shaham
Shai
S
Leroux
Michel R
MR
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
D029742:Caenorhabditis elegans Proteins; C410596:DAF-19 protein, C elegans; D014157:Transcription Factors; D049452:Green Fluorescent Proteins