c21orf59/kurly Controls Both Cilia Motility and Polarization.

Titlec21orf59/kurly Controls Both Cilia Motility and Polarization.
Publication TypeJournal Article
Year of Publication2016
AuthorsJaffe, KM, Grimes, DT, Schottenfeld-Roames, J, Werner, ME, Ku, T-SJ, Kim, SK, Pelliccia, JL, Morante, NFC, Mitchell, BJ, Burdine, RD
JournalCell Rep
Date Published2016 Mar 01
KeywordsAnimals, Binding Sites, Cell Movement, Cell Polarity, Cilia, CRISPR-Cas Systems, Dishevelled Proteins, Embryo, Nonmammalian, Gene Expression, Genetic Loci, Homologous Recombination, Kidney, Larva, LIM Domain Proteins, Membrane Proteins, Microtubules, Mutation, Protein Binding, Signal Transduction, Skin, Xenopus laevis, Xenopus Proteins, Zebrafish, Zebrafish Proteins

<p>Cilia are microtubule-based projections that function in the movement of extracellular fluid. This requires cilia to be: (1) motile and driven by dynein complexes and (2) correctly polarized on the surface of cells, which requires planar cell polarity (PCP). Few factors that regulate both processes have been discovered. We reveal that C21orf59/Kurly (Kur), a cytoplasmic protein with some enrichment at the base of cilia, is needed for motility; zebrafish mutants exhibit characteristic developmental abnormalities and dynein arm defects. kur was also required for proper cilia polarization in the zebrafish kidney and the larval skin of Xenopus laevis. CRISPR/Cas9 coupled with homologous recombination to disrupt the endogenous kur locus in Xenopus resulted in the asymmetric localization of the PCP protein Prickle2 being lost in mutant multiciliated cells. Kur also makes interactions with other PCP components, including Disheveled. This supports a model wherein Kur plays a dual role in cilia motility and polarization.</p>

Alternate JournalCell Rep
PubMed ID26904945
PubMed Central IDPMC4775428
Grant ListP30 CA060553 / CA / NCI NIH HHS / United States
2R01HD048584 / HD / NICHD NIH HHS / United States
#2R01GM089970 / GM / NIGMS NIH HHS / United States
R01 HD048584 / HD / NICHD NIH HHS / United States
F32 HD060396 / HD / NICHD NIH HHS / United States
R01 GM089970 / GM / NIGMS NIH HHS / United States
#1F32HD060396-01A1 / HD / NICHD NIH HHS / United States