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Cell Rep March 1, 2016; 14 (8): 1841-9.

c21orf59/kurly Controls Both Cilia Motility and Polarization.

Jaffe KM , Grimes DT , Schottenfeld-Roames J , Werner ME , Ku TS , Kim SK , Pelliccia JL , Morante NF , Mitchell BJ , Burdine RD .

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.

PubMed ID: 26904945
PMC ID: PMC4775428
Article link: Cell Rep
Grant support: [+]

Species referenced: Xenopus
Genes referenced: cfap298 cfap299 kidins220 myc myl7 nodal1 prickle2 rfx2 tub tuba4b vangl2
Antibodies: Tuba4b Ab4
Morpholinos: cfap298 MO1 cfap298 MO2 cfap298 MO3

Phenotypes: Xla Wt + c21orf59 MO (fig.3. o) [+]

Article Images: [+] show captions
References [+] :
Amack, Salient features of the ciliated organ of asymmetry. 2014, Pubmed