O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle control

O-GlcNAcylation of intracellular proteins is a key regulator of diverse cellular and developmental processes. Previous studies have demonstrated the acute sensitivity of cell cycle progression to chemical and genetic manipulation of O-GlcNAc homeostasis. However, the mechanisms by which O-GlcNAc reg...

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Main Authors: Conor W. Mitchell, Huijie Yuan, Marie Sønderstrup-Jensen, Andrew T. Ferenbach, Daan M. F. van Aalten
Format: Article
Language:English
Published: The Royal Society 2025-01-01
Series:Open Biology
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Online Access:https://royalsocietypublishing.org/doi/10.1098/rsob.250064
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author Conor W. Mitchell
Huijie Yuan
Marie Sønderstrup-Jensen
Andrew T. Ferenbach
Daan M. F. van Aalten
author_facet Conor W. Mitchell
Huijie Yuan
Marie Sønderstrup-Jensen
Andrew T. Ferenbach
Daan M. F. van Aalten
author_sort Conor W. Mitchell
collection DOAJ
description O-GlcNAcylation of intracellular proteins is a key regulator of diverse cellular and developmental processes. Previous studies have demonstrated the acute sensitivity of cell cycle progression to chemical and genetic manipulation of O-GlcNAc homeostasis. However, the mechanisms by which O-GlcNAc regulates the cell cycle remain poorly understood. Here, we report Ser584 O-GlcNAcylation of the RNA helicase DDX3X, a microcephaly associated protein, as a proteostatic mechanism regulating S-phase entry. Loss of Ser584 O-GlcNAcylation promoted degradation of DDX3X by the proteasome, resulting in reduced expression of the DDX3X target gene cyclin E1 and impaired cell cycle progression from G1 to S phase. These findings display how a single O-GlcNAc site affects DDX3X stability and thereby the cell cycle.
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issn 2046-2441
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publishDate 2025-01-01
publisher The Royal Society
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series Open Biology
spelling doaj-art-a868cb0a0d274d69988b1374eddcfd172025-08-20T02:44:39ZengThe Royal SocietyOpen Biology2046-24412025-01-0115710.1098/rsob.250064O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle controlConor W. Mitchell0Huijie Yuan1Marie Sønderstrup-Jensen2Andrew T. Ferenbach3Daan M. F. van Aalten4Department of Molecular Biology and Genetics, Aarhus Universitet, Aarhus, DenmarkDepartment of Molecular Biology and Genetics, Aarhus Universitet, Aarhus, DenmarkDepartment of Molecular Biology and Genetics, Aarhus Universitet, Aarhus, DenmarkDepartment of Molecular Biology and Genetics, Aarhus Universitet, Aarhus, DenmarkDepartment of Molecular Biology and Genetics, Aarhus Universitet, Aarhus, DenmarkO-GlcNAcylation of intracellular proteins is a key regulator of diverse cellular and developmental processes. Previous studies have demonstrated the acute sensitivity of cell cycle progression to chemical and genetic manipulation of O-GlcNAc homeostasis. However, the mechanisms by which O-GlcNAc regulates the cell cycle remain poorly understood. Here, we report Ser584 O-GlcNAcylation of the RNA helicase DDX3X, a microcephaly associated protein, as a proteostatic mechanism regulating S-phase entry. Loss of Ser584 O-GlcNAcylation promoted degradation of DDX3X by the proteasome, resulting in reduced expression of the DDX3X target gene cyclin E1 and impaired cell cycle progression from G1 to S phase. These findings display how a single O-GlcNAc site affects DDX3X stability and thereby the cell cycle.https://royalsocietypublishing.org/doi/10.1098/rsob.250064O-GlcNAccell cyclecell signalling
spellingShingle Conor W. Mitchell
Huijie Yuan
Marie Sønderstrup-Jensen
Andrew T. Ferenbach
Daan M. F. van Aalten
O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle control
Open Biology
O-GlcNAc
cell cycle
cell signalling
title O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle control
title_full O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle control
title_fullStr O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle control
title_full_unstemmed O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle control
title_short O-GlcNAcylation of the intellectual disability protein DDX3X exerts proteostatic cell cycle control
title_sort o glcnacylation of the intellectual disability protein ddx3x exerts proteostatic cell cycle control
topic O-GlcNAc
cell cycle
cell signalling
url https://royalsocietypublishing.org/doi/10.1098/rsob.250064
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AT mariesønderstrupjensen oglcnacylationoftheintellectualdisabilityproteinddx3xexertsproteostaticcellcyclecontrol
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