C21orf59/kurly Controls Both Cilia Motility and Polarization

Kimberly M. Jaffe, Daniel T. Grimes, Jodi Schottenfeld-Roames, Michael E. Werner, Tse Shuen J Ku, Sun K. Kim, Jose L. Pelliccia, Nicholas F C Morante, Brian J. Mitchell*, Rebecca D. Burdine

*Corresponding author for this work

Research output: Contribution to journalArticle

40 Scopus citations

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.

Original languageEnglish (US)
Pages (from-to)1841-1849
Number of pages9
JournalCell reports
Volume14
Issue number8
DOIs
StatePublished - Mar 1 2016

Keywords

  • C21orf59
  • Cilia
  • Ciliopathy
  • Disheveled
  • Kurly (Kur)
  • Multiciliated cell
  • Planar cell polarity
  • Primary ciliary dyskinesia

ASJC Scopus subject areas

  • Biochemistry, Genetics and Molecular Biology(all)

Fingerprint Dive into the research topics of 'C21orf59/kurly Controls Both Cilia Motility and Polarization'. Together they form a unique fingerprint.

  • Cite this

    Jaffe, K. M., Grimes, D. T., Schottenfeld-Roames, J., Werner, M. E., Ku, T. S. J., Kim, S. K., Pelliccia, J. L., Morante, N. F. C., Mitchell, B. J., & Burdine, R. D. (2016). C21orf59/kurly Controls Both Cilia Motility and Polarization. Cell reports, 14(8), 1841-1849. https://doi.org/10.1016/j.celrep.2016.01.069