PUBLICATION

Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control

Authors
Stevenson, N.L., Bergen, D.J.M., Xu, A., Wyatt, E., Henry, F., McCaughey, J., Vuolo, L., Hammond, C.L., Stephens, D.J.
ID
ZDB-PUB-180418-22
Date
2018
Source
Journal of Cell Science   131(9): (Journal)
Registered Authors
Bergen, Dylan, Hammond, Chrissy, Stephens, David, Stevenson, Nicola
Keywords
Calcineurin, Cilia, Giantin, Golgi, RCAN2
MeSH Terms
  • Golgi Matrix Proteins/genetics
  • Golgi Matrix Proteins/metabolism
  • Gene Knockout Techniques
  • Centrioles/genetics
  • Centrioles/metabolism*
  • Cell Cycle Proteins/metabolism*
  • Retinal Pigment Epithelium/cytology
  • Retinal Pigment Epithelium/metabolism
  • Humans
  • Cilia/genetics
  • Cilia/metabolism*
  • Muscle Proteins/genetics
  • Muscle Proteins/metabolism*
  • Zebrafish
  • Animals
(all 15)
PubMed
29643119 Full text @ J. Cell Sci.
Abstract
Almost every cell in the human body extends a primary cilium. Defective cilia function leads to a set of disorders known as ciliopathies characterised by debilitating developmental defects affecting many tissues. Here we report a new role for regulator of calcineurin 2, RCAN2, in primary cilia function. It localises to centrioles and the basal body and is required to maintain normal cilia length. RCAN2 was identified as the most strongly upregulated gene from a comparative RNAseq analysis of cells in which expression of the Golgi matrix protein giantin had been abolished by gene editing. In contrast to previous work where we showed that depletion of giantin by RNAi results in defects in ciliogenesis and in cilia length control, giantin knockout cells generate normal cilia on serum withdrawal. Furthermore, giantin knockout zebrafish show increased expression of RCAN2. Importantly, suppression of RCAN2 expression in giantin knockout cells results in the same defects in cilia length control seen on RNAi of giantin itself. Together these data define RCAN2 as a regulator of cilia function that can compensate for loss of giantin function.
Genes / Markers
Figures
Figure Gallery (1 images)
Show all Figures
Expression
Phenotype
Fish Conditions Stage Phenotype Figure
golgb1bsl077/bsl077standard conditionsDays 45-89
golgb1sa11389/sa11389standard conditionsDays 45-89
1 - 2 of 2
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
bsl077
    		Small Deletion
    sa11389
      		Point Mutation
      1 - 2 of 2
      Show
      Human Disease / Model
      No data available
      Sequence Targeting Reagents
      No data available
      Fish
      Fish
      golgb1bsl077/bsl077
      golgb1sa11389/sa11389
      WT
      1 - 3 of 3
      Show
      Antibodies
      No data available
      Orthology
      No data available
      Engineered Foreign Genes
      No data available
      Mapping
      No data available
      Your Input Welcome
      We welcome your input and comments. Please use this form to recommend updates to the information in ZFIN. We appreciate as much detail as possible and references as appropriate. We will review your comments promptly.
      Citation
      Stevenson, N.L., Bergen, D.J.M., Xu, A., Wyatt, E., Henry, F., McCaughey, J., Vuolo, L., Hammond, C.L., Stephens, D.J. (2018) Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control. Journal of Cell Science. 131(9).