PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistance

Abstract RNF168 is an E3 ubiquitin ligase critical to the mammalian DNA double-strand break repair response. The protein is recruited to and amplifies ubiquitin signals at damaged chromatin and, if not properly regulated, can drive an uncontrolled ubiquitin cascade potentially harmful to repair outc...

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Main Authors: Anoop S. Chauhan, Matthew J. W. Mackintosh, Joseph Cassar, Alexander J. Lanz, Mohammed Jamshad, Hannah L. Mackay, Alexander J. Garvin, Alexandra K. Walker, Satpal S. Jhujh, Teresa Carlomagno, Aneika C. Leney, Grant S. Stewart, Joanna R. Morris
Format: Article
Language:English
Published: Nature Portfolio 2025-04-01
Series:Nature Communications
Online Access:https://doi.org/10.1038/s41467-025-56974-9
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author Anoop S. Chauhan
Matthew J. W. Mackintosh
Joseph Cassar
Alexander J. Lanz
Mohammed Jamshad
Hannah L. Mackay
Alexander J. Garvin
Alexandra K. Walker
Satpal S. Jhujh
Teresa Carlomagno
Aneika C. Leney
Grant S. Stewart
Joanna R. Morris
author_facet Anoop S. Chauhan
Matthew J. W. Mackintosh
Joseph Cassar
Alexander J. Lanz
Mohammed Jamshad
Hannah L. Mackay
Alexander J. Garvin
Alexandra K. Walker
Satpal S. Jhujh
Teresa Carlomagno
Aneika C. Leney
Grant S. Stewart
Joanna R. Morris
author_sort Anoop S. Chauhan
collection DOAJ
description Abstract RNF168 is an E3 ubiquitin ligase critical to the mammalian DNA double-strand break repair response. The protein is recruited to and amplifies ubiquitin signals at damaged chromatin and, if not properly regulated, can drive an uncontrolled ubiquitin cascade potentially harmful to repair outcomes. Several indirect mechanisms restrict RNF168 positive feedback, and a longstanding question has been whether these alone suppress excessive RNF168 signaling or whether mechanisms to remove RNF168 from damaged chromatin exist. Here, we reveal a cascade of post-translational modifications which act at three adjacent amino acids, threonine-208, proline-209 and lysine-210, to process RNF168 actively. Phosphorylation at threonine-208 by CDK1/2 induces interaction with the peptidyl-prolyl isomerase PIN1. PIN1 promotes RNF168 SUMOylation at lysine-210, resulting in p97/VCP mediated removal. These actions promote RNF168 clearance and limit RNF168 chromatin build-up. Thus, single amino acid substitutions of the regulatory motif (SUMO-PIN1-assisted Chromatin Regulator, SPaCR) that restrict PIN1 interaction or SUMOylation are sufficient to drive supraphysiological accumulation of RNF168, increased ubiquitin signaling, excessive 53BP1 recruitment and radiosensitivity. Our findings define a mechanism of direct RNF168 regulation that is part of the normal damage response, promoting RNF168 dissociation from chromatin and limiting deleterious ubiquitin signaling.
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spelling doaj-art-3de878f1171146d4b22c78744fd8a63d2025-08-20T03:18:34ZengNature PortfolioNature Communications2041-17232025-04-0116111810.1038/s41467-025-56974-9PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistanceAnoop S. Chauhan0Matthew J. W. Mackintosh1Joseph Cassar2Alexander J. Lanz3Mohammed Jamshad4Hannah L. Mackay5Alexander J. Garvin6Alexandra K. Walker7Satpal S. Jhujh8Teresa Carlomagno9Aneika C. Leney10Grant S. Stewart11Joanna R. Morris12Department of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamBirmingham Centre for Genome Biology and Department of Cancer and Genomic Sciences, Medicine and Health, School of University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamBirmingham Centre for Genome Biology and Department of Cancer and Genomic Sciences, Medicine and Health, School of University of BirminghamBirmingham Centre for Genome Biology and Department of Cancer and Genomic Sciences, Medicine and Health, School of University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamDepartment of Cancer and Genomic Sciences, School of Medical Sciences, College of Medicine and Health, University of BirminghamAbstract RNF168 is an E3 ubiquitin ligase critical to the mammalian DNA double-strand break repair response. The protein is recruited to and amplifies ubiquitin signals at damaged chromatin and, if not properly regulated, can drive an uncontrolled ubiquitin cascade potentially harmful to repair outcomes. Several indirect mechanisms restrict RNF168 positive feedback, and a longstanding question has been whether these alone suppress excessive RNF168 signaling or whether mechanisms to remove RNF168 from damaged chromatin exist. Here, we reveal a cascade of post-translational modifications which act at three adjacent amino acids, threonine-208, proline-209 and lysine-210, to process RNF168 actively. Phosphorylation at threonine-208 by CDK1/2 induces interaction with the peptidyl-prolyl isomerase PIN1. PIN1 promotes RNF168 SUMOylation at lysine-210, resulting in p97/VCP mediated removal. These actions promote RNF168 clearance and limit RNF168 chromatin build-up. Thus, single amino acid substitutions of the regulatory motif (SUMO-PIN1-assisted Chromatin Regulator, SPaCR) that restrict PIN1 interaction or SUMOylation are sufficient to drive supraphysiological accumulation of RNF168, increased ubiquitin signaling, excessive 53BP1 recruitment and radiosensitivity. Our findings define a mechanism of direct RNF168 regulation that is part of the normal damage response, promoting RNF168 dissociation from chromatin and limiting deleterious ubiquitin signaling.https://doi.org/10.1038/s41467-025-56974-9
spellingShingle Anoop S. Chauhan
Matthew J. W. Mackintosh
Joseph Cassar
Alexander J. Lanz
Mohammed Jamshad
Hannah L. Mackay
Alexander J. Garvin
Alexandra K. Walker
Satpal S. Jhujh
Teresa Carlomagno
Aneika C. Leney
Grant S. Stewart
Joanna R. Morris
PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistance
Nature Communications
title PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistance
title_full PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistance
title_fullStr PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistance
title_full_unstemmed PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistance
title_short PIN1-SUMO2/3 motif suppresses excessive RNF168 chromatin accumulation and ubiquitin signaling to promote IR resistance
title_sort pin1 sumo2 3 motif suppresses excessive rnf168 chromatin accumulation and ubiquitin signaling to promote ir resistance
url https://doi.org/10.1038/s41467-025-56974-9
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