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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, 22 Golgi-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 both mammalian 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.

SUBMITTER: Stevenson NL 

PROVIDER: S-EPMC5769581 | biostudies-literature | 2017 Dec

REPOSITORIES: biostudies-literature

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

Stevenson Nicola L NL   Bergen Dylan J M DJM   Skinner Roderick E H REH   Kague Erika E   Martin-Silverstone Elizabeth E   Robson Brown Kate A KA   Hammond Chrissy L CL   Stephens David J DJ  

Journal of cell science 20171101 24


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, 22 Golgi-resident glycosyltransferases, but not glycan-processin  ...[more]

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