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Published online 30 October 2000. doi:10.1083/jcb.151.3.709
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© The Rockefeller University Press, 0021-9525/2000//709 $5.00
The Journal of Cell Biology, Volume 151, Number 3, , 2000 709-718

Original Article

Chlamydomonas IFT88 and Its Mouse Homologue, Polycystic Kidney Disease Gene Tg737, Are Required for Assembly of Cilia and Flagella



Gregory J. Pazoura, Bethany L. Dickerta, Yvonne Vucicab, E. Scott Seeleyb, Joel L. Rosenbaumb, George B. Witmana, and Douglas G. Colec

a Department of Cell Biology, University of Massachusetts Medical School, Worcester, Massachusetts 01655
b Department of Molecular, Cellular, and Developmental Biology, Yale University, New Haven, Connecticut 06520
c Department of Microbiology, Molecular Biology, and Biochemistry, University of Idaho, Moscow, Idaho 83844
Department of Microbiology, Molecular Biology, and Biochemistry, Life Science South 142, University of Idaho, Moscow, ID 83844-3052.208-885-6518208-885-4071

Intraflagellar transport (IFT) is a rapid movement of multi-subunit protein particles along flagellar microtubules and is required for assembly and maintenance of eukaryotic flagella. We cloned and sequenced a Chlamydomonas cDNA encoding the IFT88 subunit of the IFT particle and identified a Chlamydomonas insertional mutant that is missing this gene. The phenotype of this mutant is normal except for the complete absence of flagella. IFT88 is homologous to mouse and human genes called Tg737. Mice with defects in Tg737 die shortly after birth from polycystic kidney disease. We show that the primary cilia in the kidney of Tg737 mutant mice are shorter than normal. This indicates that IFT is important for primary cilia assembly in mammals. It is likely that primary cilia have an important function in the kidney and that defects in their assembly can lead to polycystic kidney disease.

Key Words: orpk • intraflagellar transport • primary cilia • kinesin-II • cytoplasmic dynein

© 2000 The Rockefeller University Press

Mr. Seeley's current address is Dartmouth Medical School, Department of Biochemistry, Hanover, NH 03755.

Ms. Vucica's current address is Cell Biology Group, School of Plant Science, The University of Tasmania, Hobart, Tasmania 7001, Australia.

Abbreviations used in this paper: IFT, intraflagellar transport; TPR, tetratricopeptide repeats.


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