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Giantin-knockout models reveal a feedback loop between Golgi function and glycosyltransferase expression

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Giantin-knockout models reveal a feedback loop between Golgi function and glycosyltransferase expression. / Stevenson, Nicola L.; Bergen, Dylan J.M.; Skinner, Roderick E.H.; Kague, Erika; Martin-Silverstone, Elizabeth; Robson Brown, Kate A.; Hammond, Chrissy L.; Stephens, David J.

In: Journal of Cell Science, Vol. 130, No. 24, 01.12.2017, p. 4132-4143.

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@article{5a39dba137e840e2aa6c664d6f63559d,
title = "Giantin-knockout models reveal a feedback loop between Golgi function and glycosyltransferase expression",
abstract = "The Golgi is the cellular hub for complex glycosylation, controlling accurate processing of complex proteoglycans, receptors, ligands and glycolipids. Its structure and organisation are dependent on golgins, which tether cisternal membranes and incoming transport vesicles. Here, we show that knockout of the largest golgin, giantin, leads to substantial changes in gene expression but only limited effects on Golgi structure.Notably, 22Golgi-resident glycosyltransferases, but not glycan-processing enzymes or the ER glycosylation machinery, are differentially expressed following giantin ablation. This includes near-complete loss of function of GALNT3 in bothmammalian cell and zebrafish models. Giantin-knockout zebrafish exhibit hyperostosis and ectopic calcium deposits, recapitulating phenotypes of hyperphosphatemic familial tumoral calcinosis, a disease caused by mutations in GALNT3. These data reveal a new feature of Golgi homeostasis: the ability to regulate glycosyltransferase expression to generate a functional proteoglycome.",
keywords = "GALNT3, Giantin, Glycosylation, Golgi, Hyperphosphatemic tumoral calcinosis, Zebrafish",
author = "Stevenson, {Nicola L.} and Bergen, {Dylan J.M.} and Skinner, {Roderick E.H.} and Erika Kague and Elizabeth Martin-Silverstone and {Robson Brown}, {Kate A.} and Hammond, {Chrissy L.} and Stephens, {David J.}",
note = "{\circledC} 2017. Published by The Company of Biologists Ltd.",
year = "2017",
month = "12",
day = "1",
doi = "10.1101/123547",
language = "English",
volume = "130",
pages = "4132--4143",
journal = "Journal of Cell Science",
issn = "0021-9533",
publisher = "Company of Biologists Ltd",
number = "24",

}

RIS - suitable for import to EndNote

TY - JOUR

T1 - Giantin-knockout models reveal a feedback loop between Golgi function and glycosyltransferase expression

AU - Stevenson, Nicola L.

AU - Bergen, Dylan J.M.

AU - Skinner, Roderick E.H.

AU - Kague, Erika

AU - Martin-Silverstone, Elizabeth

AU - Robson Brown, Kate A.

AU - Hammond, Chrissy L.

AU - Stephens, David J.

N1 - © 2017. Published by The Company of Biologists Ltd.

PY - 2017/12/1

Y1 - 2017/12/1

N2 - The Golgi is the cellular hub for complex glycosylation, controlling accurate processing of complex proteoglycans, receptors, ligands and glycolipids. Its structure and organisation are dependent on golgins, which tether cisternal membranes and incoming transport vesicles. Here, we show that knockout of the largest golgin, giantin, leads to substantial changes in gene expression but only limited effects on Golgi structure.Notably, 22Golgi-resident glycosyltransferases, but not glycan-processing enzymes or the ER glycosylation machinery, are differentially expressed following giantin ablation. This includes near-complete loss of function of GALNT3 in bothmammalian cell and zebrafish models. Giantin-knockout zebrafish exhibit hyperostosis and ectopic calcium deposits, recapitulating phenotypes of hyperphosphatemic familial tumoral calcinosis, a disease caused by mutations in GALNT3. These data reveal a new feature of Golgi homeostasis: the ability to regulate glycosyltransferase expression to generate a functional proteoglycome.

AB - The Golgi is the cellular hub for complex glycosylation, controlling accurate processing of complex proteoglycans, receptors, ligands and glycolipids. Its structure and organisation are dependent on golgins, which tether cisternal membranes and incoming transport vesicles. Here, we show that knockout of the largest golgin, giantin, leads to substantial changes in gene expression but only limited effects on Golgi structure.Notably, 22Golgi-resident glycosyltransferases, but not glycan-processing enzymes or the ER glycosylation machinery, are differentially expressed following giantin ablation. This includes near-complete loss of function of GALNT3 in bothmammalian cell and zebrafish models. Giantin-knockout zebrafish exhibit hyperostosis and ectopic calcium deposits, recapitulating phenotypes of hyperphosphatemic familial tumoral calcinosis, a disease caused by mutations in GALNT3. These data reveal a new feature of Golgi homeostasis: the ability to regulate glycosyltransferase expression to generate a functional proteoglycome.

KW - GALNT3

KW - Giantin

KW - Glycosylation

KW - Golgi

KW - Hyperphosphatemic tumoral calcinosis

KW - Zebrafish

UR - http://www.scopus.com/inward/record.url?scp=85038265065&partnerID=8YFLogxK

U2 - 10.1101/123547

DO - 10.1101/123547

M3 - Article

VL - 130

SP - 4132

EP - 4143

JO - Journal of Cell Science

JF - Journal of Cell Science

SN - 0021-9533

IS - 24

ER -