Identification of CHE-13, a novel intraflagellar transport protein required for cilia formation.
Cilia are present on cells of many eukaryotic organisms and recent data in the mouse suggest that ciliary defects can cause severe developmental abnormalities and disease. Studies across eukaryotic systems indicate that cilia are constructed and maintained through a highly conserved process termed intraflagellar transport (IFT), for which many of the proteins involved have yet to be identified. IFT describes the movement of large protein particles consisting of an A and a B complex along the cilia axoneme in anterograde and retrograde directions. Herein we describe a novel C. elegans gene, F59C6.7/9, that is required for cilia assembly and whose function is disrupted in che-13 ciliogenic mutants. As previously shown for all IFT complex B genes identified to date, expression of che-13 (F59C6.7/9) is regulated by the RFX-type transcription factor DAF-19, suggesting a conserved transcriptional pathway in ciliogenesis. Fluorescent-tagged CHE-13 protein concentrates at the base of cilia and moves along the axoneme as expected for an IFT protein. Furthermore, loss of che-13 differentially affects the localization of two known IFT complex B proteins, OSM-5 and OSM-6, implying that CHE-13 functions as part of this complex. Overall, our data confirm that CHE-13 is an IFT protein and further that the IFT particle assembles in an ordered process through specific protein-protein interactions.
Haycraft
Courtney J
CJ
Department of Cell Biology, University of Alabama at Birmingham Medical Center, Birmingham, AL 35294, USA.
Schafer
Jenny C
JC
Zhang
Qihong
Q
Taulman
Patrick D
PD
Yoder
Bradley K
BK
D020816:Amino Acid Motifs / Q000235:genetics; D000818:Animals; D017173:Caenorhabditis elegans / Q000166:cytology / Q000378:metabolism*; D029742:Caenorhabditis elegans Proteins / Q000235:genetics / Q000302:isolation & purification* / Q000378:metabolism; D002352:Carrier Proteins / Q000235:genetics / Q000302:isolation & purification* / Q000378:metabolism; D002451:Cell Compartmentation / Q000235:genetics; D002478:Cells, Cultured; D002923:Cilia / Q000378:metabolism*; D018076:DNA, Complementary / Q000032:analysis / Q000235:genetics; D004268:DNA-Binding Proteins; D005407:Flagella / Q000378:metabolism*; D008969:Molecular Sequence Data; D009154:Mutation / Q000235:genetics; D009419:Nerve Tissue Proteins / Q000235:genetics / Q000378:metabolism; D009475:Neurons, Afferent / Q000378:metabolism; D009479:Neuropeptides / Q000235:genetics / Q000378:metabolism; D011401:Promoter Regions, Genetic / Q000235:genetics; D021381:Protein Transport / Q000502:physiology*; D017386:Sequence Homology, Amino Acid; D012689:Sequence Homology, Nucleic Acid; D014157:Transcription Factors / Q000235:genetics / Q000378:metabolism
C474339:CHE-13 protein, C elegans; D029742:Caenorhabditis elegans Proteins; D002352:Carrier Proteins; C410596:DAF-19 protein, C elegans; D018076:DNA, Complementary; D004268:DNA-Binding Proteins; C489536:Hip1 protein, mouse; D009419:Nerve Tissue Proteins; D009479:Neuropeptides; C111073:Osm-6 protein, C elegans; D014157:Transcription Factors; C427364:osm-5 protein, C elegans