Article ; Online: Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control.
2018 Volume 131, Issue 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, which are characterised by debilitating developmental defects that affect many tissues. Here, we report a new role ... ...
| Abstract | Almost every cell in the human body extends a primary cilium. Defective cilia function leads to a set of disorders known as ciliopathies, which are characterised by debilitating developmental defects that affect 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 after 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 the control of cilia length that are seen upon RNAi of giantin itself. Together, these data define RCAN2 as a regulator of cilia function that can compensate for the loss of giantin function. |
||||||||||
|---|---|---|---|---|---|---|---|---|---|---|---|
| MeSH term(s) | Animals ; Cell Cycle Proteins/metabolism ; Centrioles/genetics ; Centrioles/metabolism ; Cilia/genetics ; Cilia/metabolism ; Gene Knockout Techniques ; Golgi Matrix Proteins/genetics ; Golgi Matrix Proteins/metabolism ; Humans ; Muscle Proteins/genetics ; Muscle Proteins/metabolism ; Retinal Pigment Epithelium/cytology ; Retinal Pigment Epithelium/metabolism ; Zebrafish | ||||||||||
| Chemical Substances | Cell Cycle Proteins ; Golgi Matrix Proteins ; Muscle Proteins ; RCAN2 protein, human ; macrogolgin | ||||||||||
| Language | English | ||||||||||
| Publishing date | 2018-05-04 | ||||||||||
| Publishing country | England | ||||||||||
| Document type | Journal Article ; Research Support, Non-U.S. Gov't | ||||||||||
| ZDB-ID | 2993-2 | ||||||||||
| ISSN | 1477-9137 ; 0021-9533 | ||||||||||
| ISSN (online) | 1477-9137 | ||||||||||
| ISSN | 0021-9533 | ||||||||||
| DOI | 10.1242/jcs.212258 | ||||||||||
| Shelf mark |
|
||||||||||
| Database | MEDical Literature Analysis and Retrieval System OnLINE |
Full text online
More links
Kategorien
In stock of ZB MED Cologne/Königswinter
| Zs.A 1200: Show issues | Location: Je nach Verfügbarkeit (siehe Angabe bei Bestand) bis Jg. 1994: Bestellungen von Artikeln über das Online-Bestellformular Jg. 1995 - 2021: Lesesall (1.OG) ab Jg. 2022: Lesesaal (EG) |
|||
| Zs.MG 14: Show issues |
Order via subito
This service is chargeable due to the Delivery terms set by subito. Orders including an article and supplementary material will be classified as separate orders. In these cases, fees will be demanded for each order.