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c21orf59/kurly controls both cilia motility and polarization #MMPMID26904945
Jaffe KM; Grimes DT; Schottenfeld-Roames J; Werner ME; Ku Ts; Kim SK; Pelliccia JL; Morante NFC; Mitchell BJ; Burdine RD
Cell Rep 2016[Mar]; 14 (8): 1841-9 PMID26904945show ga
Cilia are microtubule-based projections that function in the movement of extracellular fluid. This requires cilia to be: (1) motile, 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 multi-ciliated 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.