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

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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.

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@article{3b0e33b457ca4e7c940b90fefe3959fe,
title = "Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control",
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 defineRCAN2 as a regulator of cilia function that can compensate for the loss of giantin function.",
keywords = "Calcineurin, Cilia, Giantin, Golgi, RCAN2",
author = "Stevenson, {Nicola L.} and Bergen, {Dylan J.M.} and Amadeus Xu and Emily Wyatt and Freya Henry and Janine McCaughey and Laura Vuolo and Hammond, {Chrissy L.} and Stephens, {David J.}",
year = "2018",
month = "5",
day = "1",
doi = "10.1242/jcs.212258",
language = "English",
volume = "131",
journal = "Journal of Cell Science",
issn = "0021-9533",
publisher = "Company of Biologists Ltd",
number = "9",

}

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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 -