Chlamydomonas IFT88 and its mouse homologue, polycystic kidney disease gene tg737, are required for assembly of cilia and flagella
Pazour, Gregory J. ; Dickert, Bethany L. ; Vucica, Yvonne ; Seeley, E. Scott ; Rosenbaum, Joel L. ; Witman, George B. ; Cole, Douglas G.
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Student Authors
Faculty Advisor
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UMass Chan Affiliations
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Keywords
Animals
Chlamydomonas
Cilia
Cloning, Molecular
Conserved Sequence
Flagella
Humans
Kidney
Meiosis
Mice
Mice, Knockout
Microscopy, Electron, Scanning
Molecular Motor Proteins
Molecular Sequence Data
Mutation
Phenotype
Polycystic Kidney, Autosomal Recessive
Protein Binding
Protein Subunits
Proteins
Protozoan Proteins
Repetitive Sequences, Amino Acid
Sequence Alignment
Sequence Homology, Amino Acid
*Tumor Suppressor Proteins
Cell Biology
Physiology
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Embargo Expiration Date
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Abstract
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.
Source
J Cell Biol. 2000 Oct 30;151(3):709-18.