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
-
- Retinal Pigment Epithelium/cytology
- Retinal Pigment Epithelium/metabolism
- Golgi Matrix Proteins/genetics
- Golgi Matrix Proteins/metabolism
- Zebrafish
- Cilia/genetics
- Cilia/metabolism*
- Animals
- Humans
- Gene Knockout Techniques
- Muscle Proteins/genetics
- Muscle Proteins/metabolism*
- Cell Cycle Proteins/metabolism*
- Centrioles/genetics
- Centrioles/metabolism*
- PubMed
- 29643119 Full text @ J. Cell Sci.
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).
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
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping