USP37 prevents unscheduled replisome unloading through MCM complex deubiquitination

Abstract The CMG helicase (CDC45-MCM2-7-GINS) unwinds DNA as a component of eukaryotic replisomes. Replisome (dis)assembly is tightly coordinated with cell cycle progression to ensure genome stability. However, factors that prevent premature CMG unloading and replisome disassembly are poorly describ...

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Main Authors: Derek L. Bolhuis, Dalia Fleifel, Thomas Bonacci, Xianxi Wang, Brandon L. Mouery, Jeanette Gowen Cook, Nicholas G. Brown, Michael J. Emanuele
Format: Article
Language:English
Published: Nature Portfolio 2025-05-01
Series:Nature Communications
Online Access:https://doi.org/10.1038/s41467-025-59770-7
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author Derek L. Bolhuis
Dalia Fleifel
Thomas Bonacci
Xianxi Wang
Brandon L. Mouery
Jeanette Gowen Cook
Nicholas G. Brown
Michael J. Emanuele
author_facet Derek L. Bolhuis
Dalia Fleifel
Thomas Bonacci
Xianxi Wang
Brandon L. Mouery
Jeanette Gowen Cook
Nicholas G. Brown
Michael J. Emanuele
author_sort Derek L. Bolhuis
collection DOAJ
description Abstract The CMG helicase (CDC45-MCM2-7-GINS) unwinds DNA as a component of eukaryotic replisomes. Replisome (dis)assembly is tightly coordinated with cell cycle progression to ensure genome stability. However, factors that prevent premature CMG unloading and replisome disassembly are poorly described. Since disassembly is catalyzed by ubiquitination, deubiquitinases (DUBs) represent attractive candidates for safeguarding against untimely and deleterious CMG unloading. We combined a targeted loss-of-function screen with quantitative, single-cell analysis to identify human USP37 as a key DUB preventing replisome disassembly. We demonstrate that USP37 maintains active replisomes on S phase chromatin and promotes normal cell cycle progression. Proteomics and biochemical assays revealed USP37 interacts with the CMG complex to deubiquitinate MCM7, antagonizing replisome disassembly. Significantly, USP37 protects normal epithelial cells from oncoprotein-induced replication stress. Our findings reveal USP37 to be critical to the maintenance of replisomes in S phase and suggest USP37-targeting as a potential strategy for treating malignancies with defective DNA replication control.
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spelling doaj-art-50ae1b57bb2b4e628ef52f3d9efc62482025-08-20T02:25:16ZengNature PortfolioNature Communications2041-17232025-05-0116111710.1038/s41467-025-59770-7USP37 prevents unscheduled replisome unloading through MCM complex deubiquitinationDerek L. Bolhuis0Dalia Fleifel1Thomas Bonacci2Xianxi Wang3Brandon L. Mouery4Jeanette Gowen Cook5Nicholas G. Brown6Michael J. Emanuele7Department of Biochemistry and Biophysics and Lineberger Comprehensive Cancer Center, University of North CarolinaDepartment of Biochemistry and Biophysics and Lineberger Comprehensive Cancer Center, University of North CarolinaDepartment of Pharmacology and Lineberger Comprehensive Cancer Center, University of North CarolinaDepartment of Pharmacology and Lineberger Comprehensive Cancer Center, University of North CarolinaCurriculum in Genetics and Molecular Biology, University of North CarolinaDepartment of Biochemistry and Biophysics and Lineberger Comprehensive Cancer Center, University of North CarolinaDepartment of Pharmacology and Lineberger Comprehensive Cancer Center, University of North CarolinaDepartment of Pharmacology and Lineberger Comprehensive Cancer Center, University of North CarolinaAbstract The CMG helicase (CDC45-MCM2-7-GINS) unwinds DNA as a component of eukaryotic replisomes. Replisome (dis)assembly is tightly coordinated with cell cycle progression to ensure genome stability. However, factors that prevent premature CMG unloading and replisome disassembly are poorly described. Since disassembly is catalyzed by ubiquitination, deubiquitinases (DUBs) represent attractive candidates for safeguarding against untimely and deleterious CMG unloading. We combined a targeted loss-of-function screen with quantitative, single-cell analysis to identify human USP37 as a key DUB preventing replisome disassembly. We demonstrate that USP37 maintains active replisomes on S phase chromatin and promotes normal cell cycle progression. Proteomics and biochemical assays revealed USP37 interacts with the CMG complex to deubiquitinate MCM7, antagonizing replisome disassembly. Significantly, USP37 protects normal epithelial cells from oncoprotein-induced replication stress. Our findings reveal USP37 to be critical to the maintenance of replisomes in S phase and suggest USP37-targeting as a potential strategy for treating malignancies with defective DNA replication control.https://doi.org/10.1038/s41467-025-59770-7
spellingShingle Derek L. Bolhuis
Dalia Fleifel
Thomas Bonacci
Xianxi Wang
Brandon L. Mouery
Jeanette Gowen Cook
Nicholas G. Brown
Michael J. Emanuele
USP37 prevents unscheduled replisome unloading through MCM complex deubiquitination
Nature Communications
title USP37 prevents unscheduled replisome unloading through MCM complex deubiquitination
title_full USP37 prevents unscheduled replisome unloading through MCM complex deubiquitination
title_fullStr USP37 prevents unscheduled replisome unloading through MCM complex deubiquitination
title_full_unstemmed USP37 prevents unscheduled replisome unloading through MCM complex deubiquitination
title_short USP37 prevents unscheduled replisome unloading through MCM complex deubiquitination
title_sort usp37 prevents unscheduled replisome unloading through mcm complex deubiquitination
url https://doi.org/10.1038/s41467-025-59770-7
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