PUBLICATION
c21orf59/kurly Controls Both Cilia Motility and Polarization
- Authors
- Jaffe, K.M., Grimes, D.T., Schottenfeld-Roames, J., Werner, M.E., Ku, T.J., Kim, S.K., Pelliccia, J.L., Morante, N.F., Mitchell, B.J., Burdine, R.D.
- ID
- ZDB-PUB-160226-23
- Date
- 2016
- Source
- Cell Reports 14(8): 1841-9 (Journal)
- Registered Authors
- Burdine, Rebecca, Grimes, Daniel T., Jaffe, Kimberly
- Keywords
- Kurly (Kur), c21orf59, cilia, ciliopathy, disheveled, multiciliated cell, planar cell polarity, primary ciliary dyskinesia
- MeSH Terms
-
- Zebrafish/embryology
- Zebrafish/genetics*
- Zebrafish/metabolism
- Gene Expression
- Cell Movement
- Xenopus laevis/embryology
- Xenopus laevis/genetics*
- Xenopus laevis/metabolism
- Homologous Recombination
- Xenopus Proteins/genetics
- Xenopus Proteins/metabolism
- LIM Domain Proteins/genetics*
- LIM Domain Proteins/metabolism
- Mutation
- Animals
- Signal Transduction
- Dishevelled Proteins/genetics
- Dishevelled Proteins/metabolism
- Binding Sites
- Kidney/cytology
- Kidney/growth & development
- Kidney/metabolism
- Cell Polarity
- Microtubules/metabolism*
- Microtubules/ultrastructure
- Embryo, Nonmammalian
- Cilia/metabolism
- Protein Binding
- CRISPR-Cas Systems
- Zebrafish Proteins/genetics*
- Zebrafish Proteins/metabolism
- Genetic Loci
- Larva/genetics
- Larva/growth & development
- Larva/metabolism
- Skin/cytology
- Skin/growth & development
- Skin/metabolism
- PubMed
- 26904945 Full text @ Cell Rep.
Citation
Jaffe, K.M., Grimes, D.T., Schottenfeld-Roames, J., Werner, M.E., Ku, T.J., Kim, S.K., Pelliccia, J.L., Morante, N.F., Mitchell, B.J., Burdine, R.D. (2016) c21orf59/kurly Controls Both Cilia Motility and Polarization. Cell Reports. 14(8):1841-9.
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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping