Research output: Contribution to journal › Article
Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control. / Stevenson, Nicola L.; Bergen, Dylan J.M.; Xu, Amadeus; Wyatt, Emily; Henry, Freya; McCaughey, Janine; Vuolo, Laura; Hammond, Chrissy L.; Stephens, David J.
In: Journal of Cell Science, Vol. 131, No. 9, jcs212258, 01.05.2018.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control
AU - Stevenson, Nicola L.
AU - Bergen, Dylan J.M.
AU - Xu, Amadeus
AU - Wyatt, Emily
AU - Henry, Freya
AU - McCaughey, Janine
AU - Vuolo, Laura
AU - Hammond, Chrissy L.
AU - Stephens, David J.
PY - 2018/5/1
Y1 - 2018/5/1
N2 - 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 defineRCAN2 as a regulator of cilia function that can compensate for the loss of giantin function.
AB - 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 defineRCAN2 as a regulator of cilia function that can compensate for the loss of giantin function.
KW - Calcineurin
KW - Cilia
KW - Giantin
KW - Golgi
KW - RCAN2
UR - http://www.scopus.com/inward/record.url?scp=85046852980&partnerID=8YFLogxK
U2 - 10.1242/jcs.212258
DO - 10.1242/jcs.212258
M3 - Article
VL - 131
JO - Journal of Cell Science
JF - Journal of Cell Science
SN - 0021-9533
IS - 9
M1 - jcs212258
ER -
