Genome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphoma

Abstract Identifying tumor suppressor genes is predicted to inform on the development of novel strategies for cancer therapy. To identify new lymphoma driving processes that cooperate with oncogenic MYC, which is abnormally highly expressed in ~70% of human cancers, we use a genome-wide CRISPR gene...

Full description

Saved in:
Bibliographic Details
Main Authors: Margaret A. Potts, Shinsuke Mizutani, Yexuan Deng, Srimayee Vaidyanathan, Keziah E. Ting, Göknur Giner, Shruti Sridhar, Girija Shenoy, Yang Liao, Sarah T. Diepstraten, Andrew J. Kueh, Martin Pal, Geraldine Healey, Lin Tai, Zilu Wang, Christina König, Deeksha Kaloni, Lauren Whelan, Michael J. G. Milevskiy, Hannah D. Coughlan, Giovanna Pomilio, Andrew H. Wei, Jane E. Visvader, Anthony T. Papenfuss, Stephen Wilcox, Anand D. Jeyasekharan, Wei Shi, Emily J. Lelliott, Gemma L. Kelly, Kristin K. Brown, Andreas Strasser, Marco J. Herold
Format: Article
Language:English
Published: Nature Portfolio 2025-08-01
Series:Nature Communications
Online Access:https://doi.org/10.1038/s41467-025-62615-y
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1849226170905657344
author Margaret A. Potts
Shinsuke Mizutani
Yexuan Deng
Srimayee Vaidyanathan
Keziah E. Ting
Göknur Giner
Shruti Sridhar
Girija Shenoy
Yang Liao
Sarah T. Diepstraten
Andrew J. Kueh
Martin Pal
Geraldine Healey
Lin Tai
Zilu Wang
Christina König
Deeksha Kaloni
Lauren Whelan
Michael J. G. Milevskiy
Hannah D. Coughlan
Giovanna Pomilio
Andrew H. Wei
Jane E. Visvader
Anthony T. Papenfuss
Stephen Wilcox
Anand D. Jeyasekharan
Wei Shi
Emily J. Lelliott
Gemma L. Kelly
Kristin K. Brown
Andreas Strasser
Marco J. Herold
author_facet Margaret A. Potts
Shinsuke Mizutani
Yexuan Deng
Srimayee Vaidyanathan
Keziah E. Ting
Göknur Giner
Shruti Sridhar
Girija Shenoy
Yang Liao
Sarah T. Diepstraten
Andrew J. Kueh
Martin Pal
Geraldine Healey
Lin Tai
Zilu Wang
Christina König
Deeksha Kaloni
Lauren Whelan
Michael J. G. Milevskiy
Hannah D. Coughlan
Giovanna Pomilio
Andrew H. Wei
Jane E. Visvader
Anthony T. Papenfuss
Stephen Wilcox
Anand D. Jeyasekharan
Wei Shi
Emily J. Lelliott
Gemma L. Kelly
Kristin K. Brown
Andreas Strasser
Marco J. Herold
author_sort Margaret A. Potts
collection DOAJ
description Abstract Identifying tumor suppressor genes is predicted to inform on the development of novel strategies for cancer therapy. To identify new lymphoma driving processes that cooperate with oncogenic MYC, which is abnormally highly expressed in ~70% of human cancers, we use a genome-wide CRISPR gene knockout screen in Eµ-Myc;Cas9 transgenic hematopoietic stem and progenitor cells in vivo. We discover that loss of any of the GATOR1 complex components - NPRL3, DEPDC5, NPRL2 - significantly accelerates c-MYC-driven lymphoma development in mice. MYC-driven lymphomas lacking GATOR1 display constitutive mTOR pathway activation and are highly sensitive to mTOR inhibitors, both in vitro and in vivo. These findings identify GATOR1 suppression of mTORC1 as a tumor suppressive mechanism in MYC-driven lymphomagenesis and suggest an avenue for therapeutic intervention in GATOR1-deficient lymphomas through mTOR inhibition.
format Article
id doaj-art-e8cf85c6b6634be99a253532743f4ca5
institution Kabale University
issn 2041-1723
language English
publishDate 2025-08-01
publisher Nature Portfolio
record_format Article
series Nature Communications
spelling doaj-art-e8cf85c6b6634be99a253532743f4ca52025-08-24T11:37:08ZengNature PortfolioNature Communications2041-17232025-08-0116111410.1038/s41467-025-62615-yGenome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphomaMargaret A. Potts0Shinsuke Mizutani1Yexuan Deng2Srimayee Vaidyanathan3Keziah E. Ting4Göknur Giner5Shruti Sridhar6Girija Shenoy7Yang Liao8Sarah T. Diepstraten9Andrew J. Kueh10Martin Pal11Geraldine Healey12Lin Tai13Zilu Wang14Christina König15Deeksha Kaloni16Lauren Whelan17Michael J. G. Milevskiy18Hannah D. Coughlan19Giovanna Pomilio20Andrew H. Wei21Jane E. Visvader22Anthony T. Papenfuss23Stephen Wilcox24Anand D. Jeyasekharan25Wei Shi26Emily J. Lelliott27Gemma L. Kelly28Kristin K. Brown29Andreas Strasser30Marco J. Herold31The Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchPeter MacCallum Cancer CentrePeter MacCallum Cancer CentreThe Walter and Eliza Hall Institute of Medical ResearchDepartment of Hematology-Oncology, National University Hospital and Cancer Science Institute of Singapore, National University of SingaporeDepartment of Hematology-Oncology, National University Hospital and Cancer Science Institute of Singapore, National University of SingaporeOlivia Newton-John Cancer Research Institute, HeidelbergThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchDepartment of Hematology-Oncology, National University Hospital and Cancer Science Institute of Singapore, National University of SingaporeOlivia Newton-John Cancer Research Institute, HeidelbergThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchPeter MacCallum Cancer CentreThe Walter and Eliza Hall Institute of Medical ResearchThe Walter and Eliza Hall Institute of Medical ResearchAbstract Identifying tumor suppressor genes is predicted to inform on the development of novel strategies for cancer therapy. To identify new lymphoma driving processes that cooperate with oncogenic MYC, which is abnormally highly expressed in ~70% of human cancers, we use a genome-wide CRISPR gene knockout screen in Eµ-Myc;Cas9 transgenic hematopoietic stem and progenitor cells in vivo. We discover that loss of any of the GATOR1 complex components - NPRL3, DEPDC5, NPRL2 - significantly accelerates c-MYC-driven lymphoma development in mice. MYC-driven lymphomas lacking GATOR1 display constitutive mTOR pathway activation and are highly sensitive to mTOR inhibitors, both in vitro and in vivo. These findings identify GATOR1 suppression of mTORC1 as a tumor suppressive mechanism in MYC-driven lymphomagenesis and suggest an avenue for therapeutic intervention in GATOR1-deficient lymphomas through mTOR inhibition.https://doi.org/10.1038/s41467-025-62615-y
spellingShingle Margaret A. Potts
Shinsuke Mizutani
Yexuan Deng
Srimayee Vaidyanathan
Keziah E. Ting
Göknur Giner
Shruti Sridhar
Girija Shenoy
Yang Liao
Sarah T. Diepstraten
Andrew J. Kueh
Martin Pal
Geraldine Healey
Lin Tai
Zilu Wang
Christina König
Deeksha Kaloni
Lauren Whelan
Michael J. G. Milevskiy
Hannah D. Coughlan
Giovanna Pomilio
Andrew H. Wei
Jane E. Visvader
Anthony T. Papenfuss
Stephen Wilcox
Anand D. Jeyasekharan
Wei Shi
Emily J. Lelliott
Gemma L. Kelly
Kristin K. Brown
Andreas Strasser
Marco J. Herold
Genome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphoma
Nature Communications
title Genome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphoma
title_full Genome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphoma
title_fullStr Genome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphoma
title_full_unstemmed Genome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphoma
title_short Genome-wide in vivo CRISPR screens identify GATOR1 complex as a tumor suppressor in Myc-driven lymphoma
title_sort genome wide in vivo crispr screens identify gator1 complex as a tumor suppressor in myc driven lymphoma
url https://doi.org/10.1038/s41467-025-62615-y
work_keys_str_mv AT margaretapotts genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT shinsukemizutani genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT yexuandeng genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT srimayeevaidyanathan genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT keziaheting genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT goknurginer genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT shrutisridhar genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT girijashenoy genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT yangliao genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT sarahtdiepstraten genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT andrewjkueh genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT martinpal genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT geraldinehealey genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT lintai genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT ziluwang genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT christinakonig genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT deekshakaloni genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT laurenwhelan genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT michaeljgmilevskiy genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT hannahdcoughlan genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT giovannapomilio genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT andrewhwei genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT janeevisvader genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT anthonytpapenfuss genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT stephenwilcox genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT ananddjeyasekharan genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT weishi genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT emilyjlelliott genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT gemmalkelly genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT kristinkbrown genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT andreasstrasser genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma
AT marcojherold genomewideinvivocrisprscreensidentifygator1complexasatumorsuppressorinmycdrivenlymphoma