spacer gif spacer gif spacer gif spacer gif ARCHIVE ANNOUNCEMENT! spacer gif
QUICK SEARCH:   [advanced]


spacer gif
     Home     Help     Feedback     Subscriptions     Archive     Search     Table of Contents    

This Article
Right arrow Full Text (PDF)
Right arrow References
Right arrow Alert me when this article is cited
Right arrow Alert me if a correction is posted
Services
Right arrow Email this article to a friend
Right arrow Similar articles in this journal
Right arrow Similar articles in PubMed
Right arrow Alert me to new issues of the journal
Right arrow Download to citation manager
Right arrow reprints & permissions
Citing Articles
Right arrow Citing Articles via HighWire
Right arrow Citing Articles via Google Scholar
Google Scholar
Right arrow Articles by Haycraft, C. J.
Right arrow Articles by Yoder, B. K.
Right arrow Search for Related Content
PubMed
Right arrow PubMed Citation
Right arrow Articles by Haycraft, C. J.
Right arrow Articles by Yoder, B. K.

Development, Vol 128, Issue 9 1493-1505, Copyright © 2001 by Company of Biologists

JOURNAL ARTICLES

The C. elegans homolog of the murine cystic kidney disease gene Tg737 functions in a ciliogenic pathway and is disrupted in osm-5 mutant worms

CJ Haycraft, P Swoboda, PD Taulman, JH Thomas and BK Yoder
Department of Cell Biology, University of Alabama at Birmingham Medical Center, Birmingham, AL 35294, USA.

Cilia and flagella are important organelles involved in diverse functions such as fluid and cell movement, sensory perception and embryonic patterning. They are devoid of protein synthesis, thus their formation and maintenance requires the movement of protein complexes from the cytoplasm into the cilium and flagellum axoneme by intraflagellar transport (IFT), a conserved process common to all ciliated or flagellated eukaryotic cells. We report that mutations in the Caenorhabditis elegans gene Y41g9a.1 are responsible for the ciliary defects in osm-5 mutant worms. This was confirmed by transgenic rescue of osm-5(p813) mutants using the wild-type Y41g9a.1 gene. osm-5 encodes a tetratricopeptide repeat (TPR)-containing protein that is the homolog of murine polaris (Tg737), a protein associated with cystic kidney disease and left-right axis patterning defects in the mouse. osm-5 is expressed in ciliated sensory neurons in C. elegans and its expression is regulated by DAF-19, an RFX-type transcription factor that governs the expression of other genes involved in cilia formation in the worm. Similar to murine polaris, the OSM-5 protein was found to concentrate at the cilium base and within the cilium axoneme as shown by an OSM-5::GFP translational fusion and immunofluorescence. Furthermore, time-lapse imaging of OSM-5::GFP fusion protein shows fluorescent particle migration within the cilia. Overall, the data support a crucial role for osm-5 in a conserved ciliogenic pathway, most likely as a component of the IFT process. http://www.biologists.com/Development/movies/dev3342.html


This article has been cited by other articles:


Home page
 Mol. Biol. CellHome page
J. C. Hoeng, S. C. Dawson, S. A. House, M. S. Sagolla, J. K. Pham, J. J. Mancuso, J. Lowe, and W. Z. Cande
High-Resolution Crystal Structure and In Vivo Function of a Kinesin-2 Homologue in Giardia intestinalis
Mol. Biol. Cell, July 1, 2008; 19(7): 3124 - 3137.
[Abstract] [Full Text] [PDF]

Home page
 J. Cell Sci.Home page
B. L. Krock and B. D. Perkins
The intraflagellar transport protein IFT57 is required for cilia maintenance and regulates IFT-particle-kinesin-II dissociation in vertebrate photoreceptors
J. Cell Sci., June 1, 2008; 121(11): 1907 - 1915.
[Abstract] [Full Text] [PDF]

Home page
 Mol. Biol. CellHome page
C. L. Williams, M. E. Winkelbauer, J. C. Schafer, E. J. Michaud, and B. K. Yoder
Functional Redundancy of the B9 Proteins and Nephrocystins in Caenorhabditis elegans Ciliogenesis
Mol. Biol. Cell, May 1, 2008; 19(5): 2154 - 2168.
[Abstract] [Full Text] [PDF]

Home page
 Mol. Biol. CellHome page
C.-C. Tsao and M. A. Gorovsky
Different Effects of Tetrahymena IFT172 Domains on Anterograde and Retrograde Intraflagellar Transport
Mol. Biol. Cell, April 1, 2008; 19(4): 1450 - 1461.
[Abstract] [Full Text] [PDF]

Home page
 GeneticsHome page
T. Bacaj, Y. Lu, and S. Shaham
The Conserved Proteins CHE-12 and DYF-11 Are Required for Sensory Cilium Function in Caenorhabditis elegans
Genetics, February 1, 2008; 178(2): 989 - 1002.
[Abstract] [Full Text] [PDF]

Home page
 Physiol. GenomicsHome page
T. S. McClintock, C. E. Glasser, S. C. Bose, and D. A. Bergman
Tissue expression patterns identify mouse cilia genes
Physiol Genomics, January 17, 2008; 32(2): 198 - 206.
[Abstract] [Full Text] [PDF]

Home page
 GENES CELLSHome page
H. Kunitomo and Y. Iino
Caenorhabditis elegans DYF-11, an orthologue of mammalian Traf3ip1/MIP-T3, is required for sensory cilia formation.
Genes Cells, January 1, 2008; 13(1): 13 - 25.
[Abstract] [Full Text] [PDF]

Home page
 J. Neurosci.Home page
C. V. Gabel, H. Gabel, D. Pavlichin, A. Kao, D. A. Clark, and A. D. T. Samuel
Neural Circuits Mediate Electrosensory Behavior in Caenorhabditis elegans
J. Neurosci., July 11, 2007; 27(28): 7586 - 7596.
[Abstract] [Full Text] [PDF]

Home page
 J. Am. Soc. Nephrol.Home page
B. K. Yoder
Role of Primary Cilia in the Pathogenesis of Polycystic Kidney Disease
J. Am. Soc. Nephrol., May 1, 2007; 18(5): 1381 - 1388.
[Abstract] [Full Text] [PDF]

Home page
 Proc. Natl. Acad. Sci. USAHome page
J. Burghoorn, M. P. J. Dekkers, S. Rademakers, T. de Jong, R. Willemsen, and G. Jansen
Mutation of the MAP kinase DYF-5 affects docking and undocking of kinesin-2 motors and reduces their speed in the cilia of Caenorhabditis elegans
PNAS, April 24, 2007; 104(17): 7157 - 7162.
[Abstract] [Full Text] [PDF]

Home page
 DiabetesHome page
A. Ait-Lounis, D. Baas, E. Barras, C. Benadiba, A. Charollais, R. Nlend Nlend, D. Liegeois, P. Meda, B. Durand, and W. Reith
Novel Function of the Ciliogenic Transcription Factor RFX3 in Development of the Endocrine Pancreas
Diabetes, April 1, 2007; 56(4): 950 - 959.
[Abstract] [Full Text] [PDF]

Home page
 Am. J. Physiol. Cell Physiol.Home page
B. Banizs, P. Komlosi, M. O. Bevensee, E. M. Schwiebert, P. D. Bell, and B. K. Yoder
Altered pHi regulation and Na+/HCO3- transporter activity in choroid plexus of cilia-defective Tg737orpk mutant mouse
Am J Physiol Cell Physiol, April 1, 2007; 292(4): C1409 - C1416.
[Abstract] [Full Text] [PDF]

Home page
 DevelopmentHome page
Y. Liu, N. Pathak, A. Kramer-Zucker, and I. A. Drummond
Notch signaling controls the differentiation of transporting epithelia and multiciliated cells in the zebrafish pronephros
Development, March 15, 2007; 134(6): 1111 - 1122.
[Abstract] [Full Text] [PDF]

Home page
 J. Cell Sci.Home page
A. Robert, G. Margall-Ducos, J.-E. Guidotti, O. Bregerie, C. Celati, C. Brechot, and C. Desdouets
The intraflagellar transport component IFT88/polaris is a centrosomal protein regulating G1-S transition in non-ciliated cells
J. Cell Sci., February 15, 2007; 120(4): 628 - 637.
[Abstract] [Full Text] [PDF]

Home page
 Mol. Biol. CellHome page
O. E. Blacque, C. Li, P. N. Inglis, M. A. Esmail, G. Ou, A. K. Mah, D. L. Baillie, J. M. Scholey, and M. R. Leroux
The WD Repeat-containing Protein IFTA-1 Is Required for Retrograde Intraflagellar Transport
Mol. Biol. Cell, December 1, 2006; 17(12): 5053 - 5062.
[Abstract] [Full Text] [PDF]

Home page
 DevelopmentHome page
B. W. Bisgrove and H. J. Yost
The roles of cilia in developmental disorders and disease
Development, November 1, 2006; 133(21): 4131 - 4143.
[Abstract] [Full Text] [PDF]

Home page
 Mol. Biol. CellHome page
E. Efimenko, O. E. Blacque, G. Ou, C. J. Haycraft, B. K. Yoder, J. M. Scholey, M. R. Leroux, and P. Swoboda
Caenorhabditis elegans DYF-2, an Orthologue of Human WDR19, Is a Component of the Intraflagellar Transport Machinery in Sensory Cilia
Mol. Biol. Cell, November 1, 2006; 17(11): 4801 - 4811.
[Abstract] [Full Text] [PDF]

Home page
 J. Cell Sci.Home page
J. C. Schafer, M. E. Winkelbauer, C. L. Williams, C. J. Haycraft, R. A. Desmond, and B. K. Yoder
IFTA-2 is a conserved cilia protein involved in pathways regulating longevity and dauer formation in Caenorhabditis elegans
J. Cell Sci., October 1, 2006; 119(19): 4088 - 4100.
[Abstract] [Full Text] [PDF]

Home page
 Am. J. Pathol.Home page
J. J. Schrick, P. Vogel, A. Abuin, B. Hampton, and D. S. Rice
ADP-Ribosylation Factor-Like 3 Is Involved in Kidney and Photoreceptor Development
Am. J. Pathol., April 1, 2006; 168(4): 1288 - 1298.
[Abstract] [Full Text] [PDF]

Home page
 Am. J. Physiol. Cell Physiol.Home page
D. Olteanu, B. K. Yoder, W. Liu, M. J. Croyle, E. A. Welty, K. Rosborough, J. M. Wyss, P. D. Bell, L. M. Guay-Woodford, M. O. Bevensee, et al.
Heightened epithelial Na+ channel-mediated Na+ absorption in a murine polycystic kidney disease model epithelium lacking apical monocilia
Am J Physiol Cell Physiol, April 1, 2006; 290(4): C952 - C963.
[Abstract] [Full Text] [PDF]

Home page
 DevelopmentHome page
B. Banizs, M. M. Pike, C. L. Millican, W. B. Ferguson, P. Komlosi, J. Sheetz, P. D. Bell, E. M. Schwiebert, and B. K. Yoder
Dysfunctional cilia lead to altered ependyma and choroid plexus function, and result in the formation of hydrocephalus
Development, December 1, 2005; 132(23): 5329 - 5339.
[Abstract] [Full Text] [PDF]

Home page
 Am. J. Physiol. Renal Physiol.Home page
J. R. Davenport and B. K. Yoder
An incredible decade for the primary cilium: a look at a once-forgotten organelle
Am J Physiol Renal Physiol, December 1, 2005; 289(6): F1159 - F1169.
[Abstract] [Full Text] [PDF]

Home page
 J. Cell Sci.Home page
M. E. Winkelbauer, J. C. Schafer, C. J. Haycraft, P. Swoboda, and B. K. Yoder
The C. elegans homologs of nephrocystin-1 and nephrocystin-4 are cilia transition zone proteins involved in chemosensory perception
J. Cell Sci., December 1, 2005; 118(23): 5575 - 5587.
[Abstract] [Full Text] [PDF]

Home page
 Am. J. Physiol. Renal Physiol.Home page
W. Liu, N. S. Murcia, Y. Duan, S. Weinbaum, B. K. Yoder, E. Schwiebert, and L. M. Satlin
Mechanoregulation of intracellular Ca2+ concentration is attenuated in collecting duct of monocilium-impaired orpk mice
Am J Physiol Renal Physiol, November 1, 2005; 289(5): F978 - F988.
[Abstract] [Full Text] [PDF]

Home page
 DevelopmentHome page
E. Efimenko, K. Bubb, H. Y. Mak, T. Holzman, M. R. Leroux, G. Ruvkun, J. H. Thomas, and P. Swoboda
Analysis of xbx genes in C. elegans
Development, April 15, 2005; 132(8): 1923 - 1934.
[Abstract] [Full Text] [PDF]

Home page
 J. Am. Soc. Nephrol.Home page
M. T.F. Wolf, J. Lee, F. Panther, E. A. Otto, K.-L. Guan, and F. Hildebrandt
Expression and Phenotype Analysis of the Nephrocystin-1 and Nephrocystin-4 Homologs in Caenorhabditis elegans
J. Am. Soc. Nephrol., March 1, 2005; 16(3): 676 - 687.
[Abstract] [Full Text] [PDF]

Home page
 J. Am. Soc. Nephrol.Home page
M. M. Barr
Caenorhabditis elegans as a Model to Study Renal Development and Disease: Sexy Cilia
J. Am. Soc. Nephrol., February 1, 2005; 16(2): 305 - 312.
[Abstract] [Full Text] [PDF]

Home page
 J. Am. Soc. Nephrol.Home page
G. J. Pazour
Intraflagellar Transport and Cilia-Dependent Renal Disease: The Ciliary Hypothesis of Polycystic Kidney Disease
J. Am. Soc. Nephrol., October 1, 2004; 15(10): 2528 - 2536.
[Abstract] [Full Text] [PDF]

Home page
 PhysiologyHome page
Q. Zhang, P. D. Taulman, and B. K. Yoder
Cystic Kidney Diseases: All Roads Lead to the Cilium
Physiology, August 1, 2004; 19(4): 225 - 230.
[Abstract] [Full Text] [PDF]

Home page
 DevelopmentHome page
J. D. Baker, S. Adhikarakunnathu, and M. J. Kernan
Mechanosensory-defective, male-sterile unc mutants identify a novel basal body protein required for ciliogenesis in Drosophila
Development, July 15, 2004; 131(14): 3411 - 3422.
[Abstract] [Full Text] [PDF]

Home page
 Genes Dev.Home page
O. E. Blacque, M. J. Reardon, C. Li, J. McCarthy, M. R. Mahjoub, S. J. Ansley, J. L. Badano, A. K. Mah, P. L. Beales, W. S. Davidson, et al.
Loss of C. elegans BBS-7 and BBS-8 protein function results in cilia defects and compromised intraflagellar transport
Genes & Dev., July 1, 2004; 18(13): 1630 - 1642.
[Abstract] [Full Text] [PDF]

Home page
 Am. J. Physiol. Renal Physiol.Home page
H. F. Cantiello
Regulation of calcium signaling by polycystin-2
Am J Physiol Renal Physiol, June 1, 2004; 286(6): F1012 - F1029.
[Abstract] [Full Text] [PDF]

Home page
 Mol. Cell. Biol.Home page
E. Bonnafe, M. Touka, A. AitLounis, D. Baas, E. Barras, C. Ucla, A. Moreau, F. Flamant, R. Dubruille, P. Couble, et al.
The Transcription Factor RFX3 Directs Nodal Cilium Development and Left-Right Asymmetry Specification
Mol. Cell. Biol., May 15, 2004; 24(10): 4417 - 4427.
[Abstract] [Full Text] [PDF]

Home page
 J. Biol. Chem.Home page
Z. Gao, E. Joseph, D. M. Ruden, and X. Lu
Drosophila Pkd2 Is Haploid-insufficient for Mediating Optimal Smooth Muscle Contractility
J. Biol. Chem., April 2, 2004; 279(14): 14225 - 14231.
[Abstract] [Full Text] [PDF]

Home page
 DevelopmentHome page
H. Yu, R. F. Pretot, T. R. Burglin, and P. W. Sternberg
Distinct roles of transcription factors EGL-46 and DAF-19 in specifying the functionality of a polycystin-expressing sensory neuron necessary for C. elegans male vulva location behavior
Development, November 1, 2003; 130(21): 5217 - 5227.
[Abstract] [Full Text] [PDF]

Home page
 J. Biol. Chem.Home page
S. A. Baker, K. Freeman, K. Luby-Phelps, G. J. Pazour, and J. C. Besharse
IFT20 Links Kinesin II with a Mammalian Intraflagellar Transport Complex That Is Conserved in Motile Flagella and Sensory Cilia
J. Biol. Chem., September 5, 2003; 278(36): 34211 - 34218.
[Abstract] [Full Text] [PDF]

Home page
 Nephrol Dial TransplantHome page
D. Joly, A. Hummel, A. Ruello, and B. Knebelmann
Ciliary function of polycystins: a new model for cystogenesis
Nephrol. Dial. Transplant., September 1, 2003; 18(9): 1689 - 1692.
[Full Text] [PDF]

Home page
 J. Cell Sci.Home page
J. Pan and W. J. Snell
Kinesin II and regulated intraflagellar transport of Chlamydomonas aurora protein kinase
J. Cell Sci., June 1, 2003; 116(11): 2179 - 2186.
[Abstract] [Full Text] [PDF]

Home page
 Proc. Natl. Acad. Sci. USAHome page
J. P. Calvet
New insights into ciliary function: Kidney cysts and photoreceptors
PNAS, May 13, 2003; 100(10): 5583 - 5585.
[Full Text] [PDF]

Home page
 DevelopmentHome page
D. Watanabe, Y. Saijoh, S. Nonaka, G. Sasaki, Y. Ikawa, T. Yokoyama, and H. Hamada
The left-right determinant Inversin is a component of node monocilia and other 9+0 cilia
Development, May 1, 2003; 130(9): 1725 - 1734.
[Abstract] [Full Text] [PDF]

Home page
 Mol. Biol. CellHome page
J. C. Schafer, C. J. Haycraft, J. H. Thomas, B. K. Yoder, and P. Swoboda
XBX-1 Encodes a Dynein Light Intermediate Chain Required for Retrograde Intraflagellar Transport and Cilia Assembly in Caenorhabditis elegans
Mol. Biol. Cell, May 1, 2003; 14(5): 2057 - 2070.
[Abstract] [Full Text] [PDF]

Home page
 Genes Dev.Home page
C. J. Tabin and K. J. Vogan
A two-cilia model for vertebrate left-right axis specification
Genes & Dev., January 1, 2003; 17(1): 1 - 6.
[Full Text] [PDF]

Home page
 J. Am. Soc. Nephrol.Home page
B. K. Yoder, X. Hou, and L. M. Guay-Woodford
The Polycystic Kidney Disease Proteins, Polycystin-1, Polycystin-2, Polaris, and Cystin, Are Co-Localized in Renal Cilia
J. Am. Soc. Nephrol., October 1, 2002; 13(10): 2508 - 2516.
[Abstract] [Full Text] [PDF]

Home page
 J. Am. Soc. Nephrol.Home page
J. P. Calvet
Cilia in PKD--Letting It All Hang Out
J. Am. Soc. Nephrol., October 1, 2002; 13(10): 2614 - 2616.
[Full Text] [PDF]

Home page
 J. Am. Soc. Nephrol.Home page
P. Igarashi and S. Somlo
Genetics and Pathogenesis of Polycystic Kidney Disease
J. Am. Soc. Nephrol., September 1, 2002; 13(9): 2384 - 2398.
[Full Text] [PDF]

Home page
 Mol. Cell. ProteomicsHome page
L. E. Ostrowski, K. Blackburn, K. M. Radde, M. B. Moyer, D. M. Schlatzer, A. Moseley, and R. C. Boucher
A Proteomic Analysis of Human Cilia: Identification of Novel Components
Mol. Cell. Proteomics, June 1, 2002; 1(6): 451 - 465.
[Abstract] [Full Text] [PDF]

Home page
 J. Cell Biol.Home page
G. J. Pazour, S. A. Baker, J. A. Deane, D. G. Cole, B. L. Dickert, J. L. Rosenbaum, G. B. Witman, and J. C. Besharse
The intraflagellar transport protein, IFT88, is essential for vertebrate photoreceptor assembly and maintenance
J. Cell Biol., April 1, 2002; 157(1): 103 - 114.
[Abstract] [Full Text] [PDF]

Home page
 Am. J. Physiol. Renal Physiol.Home page
B. K. Yoder, A. Tousson, L. Millican, J. H. Wu, C. E. Bugg Jr., J. A. Schafer, and D. F. Balkovetz
Polaris, a protein disrupted in orpk mutant mice, is required for assembly of renal cilium
Am J Physiol Renal Physiol, March 1, 2002; 282(3): F541 - F552.
[Abstract] [Full Text] [PDF]

Home page
 DevelopmentHome page
R. Dubruille, A. Laurencon, C. Vandaele, E. Shishido, M. Coulon-Bublex, P. Swoboda, P. Couble, M. Kernan, and B. Durand
Drosophila Regulatory factor X is necessary for ciliated sensory neuron differentiation
Development, January 12, 2002; 129(23): 5487 - 5498.
[Abstract] [Full Text] [PDF]

Home page
 J. Cell Biol.Home page
G. J. Pazour, S. A. Baker, J. A. Deane, D. G. Cole, B. L. Dickert, J. L. Rosenbaum, G. B. Witman, and J. C. Besharse
The intraflagellar transport protein, IFT88, is essential for vertebrate photoreceptor assembly and maintenance
J. Cell Biol., April 1, 2002; 157(1): 103 - 114.
[Abstract] [Full Text] [PDF]

Home page
 Mol. Biol. CellHome page
J. Pan and W. J. Snell
Kinesin-II Is Required for Flagellar Sensory Transduction during Fertilization in Chlamydomonas
Mol. Biol. Cell, April 1, 2002; 13(4): 1417 - 1426.
[Abstract] [Full Text] [PDF]


© The Company of Biologists Ltd 2001