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The Golgi matrix protein giantin is required for normal cilia function in zebrafish

Research output: Contribution to journalArticle

Original languageEnglish
Pages (from-to)1180-1189
Number of pages10
JournalBiology Open
Volume6
Issue number8
Early online date25 May 2017
DOIs
DateAccepted/In press - 22 May 2017
DateE-pub ahead of print - 25 May 2017
DatePublished (current) - 15 Aug 2017

Abstract

The Golgi is essential for glycosylation of newly synthesised proteins including almost all cell-surface and extracellular matrix proteoglycans. Giantin, encoded by the golgb1 gene, is a member of the golgin family of proteins that reside within the Golgi stack but its function remains elusive. Loss-of-function of giantin in rats causes osteochondrodysplasia; knockout mice show milder defects, notably a cleft palate. In vitro, giantin has been implicated in Golgi organisation, biosynthetic trafficking, and ciliogenesis. Here we show that loss-of-function of giantin in zebrafish, using either morpholino or knockout techniques, causes defects in cilia function. Giantin morphants have fewer cilia in the neural tube and those remaining are longer. Mutants have the same number of cilia in the neural tube but these cilia are also elongated. Scanning electron microscopy shows that loss of giantin results in an accumulation of material at the ciliary tip, consistent with a loss-of-function of retrograde intraflagellar transport. Mutants show milder defects than morphants consistent with adaptation to loss of giantin.

    Research areas

  • Golgi, Golgi matrix, cilia, zebrafish

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    Rights statement: This is the final published version of the article (version of record). It first appeared online via Company of Biologists at http://bio.biologists.org/content/early/2017/05/24/bio.025502. Please refer to any applicable terms of use of the publisher.

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