@article{76945f8aa29b436aa0c06b0730a6886d,
title = "C21orf59/kurly Controls Both Cilia Motility and Polarization",
abstract = "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.",
keywords = "C21orf59, Cilia, Ciliopathy, Disheveled, Kurly (Kur), Multiciliated cell, Planar cell polarity, Primary ciliary dyskinesia",
author = "Jaffe, {Kimberly M.} and Grimes, {Daniel T.} and Jodi Schottenfeld-Roames and Werner, {Michael E.} and Ku, {Tse Shuen J.} and Kim, {Sun K.} and Pelliccia, {Jose L.} and Morante, {Nicholas F.C.} and Mitchell, {Brian J.} and Burdine, {Rebecca D.}",
note = "Funding Information: The authors thank: Derrick Bosco, Cori Hasty, Phillip Johnson, Heather McAllister, and LAR staff for zebrafish care; Ray Habas for Dvl constructs; K. Horvath for Renilla constructs; and Dr. Gary Laevsky and the Molecular Biology Confocal Microscopy Facility, which is a Nikon Center of Excellence; and the National Xenopus Resource Centre for animals and expertise. This research was supported in part by a competitive research award to B.J.M. from the Eugene and Millicent Bell Fellowship Fund in Tissue Engineering and the Laura and Arthur Colwin Endowed Summer Research Fellowship Fund at the Marine Biological Laboratory in Woods Hole. The research was also supported by an NRSA grant to K.M.J. (#1F32HD060396-01A1), an NIH-NIGMS grant to B.J.M. (#2R01GM089970), and an NIH-NICHD grant to R.D.B. (#2R01HD048584). Funding Information: The authors thank: Derrick Bosco, Cori Hasty, Phillip Johnson, Heather McAllister, and LAR staff for zebrafish care; Ray Habas for Dvl constructs; K. Horvath for Renilla constructs; and Dr. Gary Laevsky and the Molecular Biology Confocal Microscopy Facility, which is a Nikon Center of Excellence; and the National Xenopus Resource Centre for animals and expertise. This research was supported in part by a competitive research award to B.J.M. from the Eugene and Millicent Bell Fellowship Fund in Tissue Engineering and the Laura and Arthur Colwin Endowed Summer Research Fellowship Fund at the Marine Biological Laboratory in Woods Hole. The research was also supported by an NRSA grant to K.M.J. (#1F32HD060396-01A1), an NIH-NIGMS grant to B.J.M. (#2R01GM089970), and an NIH-NICHD grant to R.D.B. (#2R01HD048584). Publisher Copyright: {\textcopyright} 2016 The Authors.",
year = "2016",
month = mar,
day = "1",
doi = "10.1016/j.celrep.2016.01.069",
language = "English (US)",
volume = "14",
pages = "1841--1849",
journal = "Cell Reports",
issn = "2211-1247",
publisher = "Cell Press",
number = "8",
}