ArticleNature neuroscience2019
RPS25 is required for efficient RAN translation of C9orf72 and other neurodegenerative disease-associated nucleotide repeats.
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Article in Nature neuroscience, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 66 papers.
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66citing papers in PubMed
11.6field-weighted citation impact, top 1% of its field
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66 citing papers in PubMed, 109 citations in OpenAlex.
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- Microglial Dysfunction Induced by C9ORF72 Dipeptide Repeat Proteins: Biomarker and Therapeutic Perspectives.International journal of molecular sciences · 2026Review
- eVGeMdb: a manually curated database for experimentally validated genetic modifiers of neurodegenerative disorders.NAR molecular medicine · 2026Article
- A distal promoter and aberrant splicing enable canonical translation of out-of-frame proteins in Huntington's disease.bioRxiv : the preprint server for biology · 2025Article
- A comprehensive PDCoV-host proteome interaction map reveals potential antiviral targets.PLoS pathogens · 2025Article
- Machine learning-based proteomics profiling of ALS identifies downregulation of RPS29 that maintains protein homeostasis and STMN2 level.Communications biology · 2025Article
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- Ribosome Structural Changes Dynamically Affect Ribosome Function.International journal of molecular sciences · 2024Review
- Ribosomal quality control factors inhibit repeat-associated non-AUG translation from GC-rich repeats.Nucleic acids research · 2024Article
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- CAG repeat expansions create splicing acceptor sites and produce aberrant repeat-containing RNAs.Molecular cell · 2024Article
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- CLIP-Seq analysis enables the design of protective ribosomal RNA bait oligonucleotides againstScience advances · 2023Article
- CAG repeat expansions create splicing acceptor sites and produce aberrant repeat-containing RNAs.bioRxiv : the preprint server for biology · 2023Article
- Translation of dipeptide repeat proteins ineLife · 2023Article
- Article
6 more citing papers are in PubMed but not listed here.
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Authors and funding
20 authors at 6 institutions in 2 countries.
Shizuka B YamadaDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.
Tania F GendronDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL, USA.ORCID http://orcid.org/0000-0002-7335-2627
Teresa NiccoliDepartment of Neurodegenerative Disease, UCL Institute of Neurology, London, UK.
Naomi R GenuthDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.
Rosslyn GroselyDepartment of Structural Biology, Stanford University School of Medicine, Stanford, CA, USA.
Yingxiao ShiDepartment of Stem Cell Biology and Regenerative Medicine, Eli and Edythe Broad Center for Regenerative Medicine and Stem Cell Research, University of Southern California, Los Angeles, CA, USA.
Idoia GlariaDepartment of Neurodegenerative Disease, UCL Institute of Neurology, London, UK.
Nicholas J KramerDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.ORCID http://orcid.org/0000-0003-4557-8343
Lisa NakayamaDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.
Shirleen FangDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.
Tai J I DingerDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.
Annora ThoengDepartment of Neurodegenerative Disease, UCL Institute of Neurology, London, UK.
Gabriel RochaDepartment of Stem Cell Biology and Regenerative Medicine, Eli and Edythe Broad Center for Regenerative Medicine and Stem Cell Research, University of Southern California, Los Angeles, CA, USA.
Maria BarnaDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.
Joseph D PuglisiDepartment of Structural Biology, Stanford University School of Medicine, Stanford, CA, USA.
Linda PartridgeDepartment of Genetics, Evolution and Environment, Institute of Healthy Ageing, University College London, London, UK.ORCID http://orcid.org/0000-0001-9615-0094
Justin K IchidaDepartment of Stem Cell Biology and Regenerative Medicine, Eli and Edythe Broad Center for Regenerative Medicine and Stem Cell Research, University of Southern California, Los Angeles, CA, USA.
Adrian M IsaacsDepartment of Neurodegenerative Disease, UCL Institute of Neurology, London, UK.ORCID http://orcid.org/0000-0002-6820-5534
Leonard PetrucelliDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL, USA.
Aaron D GitlerDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA. agitler@stanford.edu.ORCID http://orcid.org/0000-0001-8603-1526
Stanford University · USUK Dementia Research Institute · GBUniversity of Southern California · USMayo Clinic in Florida · USStanford Medicine · USUniversity College London · GB
Funding
INSTITUTIONAL TRAINING GRANT IN GENOME SCIENCET32HG000044 · NHGRI · STANFORD UNIVERSITY · PI MICHAEL P. SNYDER · 1995 to 2026
$32.2MExpanding insights into FTD disease mechanismsR35NS097273 · NINDS · MAYO CLINIC JACKSONVILLE · PI PETRUCELLI, LEONARD · 2017 to 2024
$10.6MInnovating Yeast and Human Genetics Approaches to Define Mechanisms of Neurodegenerative DiseaseR35NS097263 · NINDS · STANFORD UNIVERSITY · PI GITLER, AARON D. · 2017 to 2024
$8.6MValidation of r(G4C2)exp-binding small molecules in vivo and c9FTD/ALS biomarker developmentP01NS099114 · NINDS · UNIVERSITY OF FLORIDA · PI DISNEY, MATTHEW D · 2017 to 2021
$6.5MValidating Secretory Autophagy as a Therapeutic Strategy for Diverse Forms of ALS and FTDR01NS097850 · NINDS · UNIVERSITY OF SOUTHERN CALIFORNIA · PI ICHIDA, JUSTIN KAWIKA · 2016 to 2024
$5.5MSpecialized Translational Control of Stem Cell Differentiation and Embryonic DevelopmentR01HD086634 · NICHD · STANFORD UNIVERSITY · PI BARNA, MARIA · 2016 to 2025
$5.5MDynamics of eukaryotic translation initiation and its controlR01GM113078 · NIGMS · STANFORD UNIVERSITY · PI PUGLISI, JOSEPH D · 2016 to 2021
$2.9MModulation of internal ribosome entry by ribosomal protein RPS25R01AI099506 · NIAID · STANFORD UNIVERSITY · PI PUGLISI, JOSEPH D, SARNOW, PETER · 2014 to 2018
$2.9MDefining modifiers and mechanisms of RAN translationRF1AG064690 · NIA · STANFORD UNIVERSITY · PI GITLER, AARON D., PUGLISI, JOSEPH D · 2019 to 2019
$2.8MDefining modifiers and mechanisms of RAN translationR01AG064690 · NIA · STANFORD UNIVERSITY · PI Aaron D. Gitler, JOSEPH D PUGLISI · 2024 to 2026
$2.3MMotor Neurone Disease Association ISAACS/APR13/818-791Motor Neurone Disease Association ISAACS/APR15/834-791NHGRI NIH HHS T32 HG000044NIAID NIH HHS R01 AI099506NIA NIH HHS R01 AG064690NIA NIH HHS RF1 AG064690NICHD NIH HHS R01 HD086634NIGMS NIH HHS R01 GM113078NINDS NIH HHS P01 NS099114NINDS NIH HHS R01 NS097850NINDS NIH HHS R35 NS097263NINDS NIH HHS R35 NS097273
Abstract
Nucleotide repeat expansions in the C9orf72 gene are the most common cause of amyotrophic lateral sclerosis and frontotemporal dementia. Unconventional translation (RAN translation) of C9orf72 repeats generates dipeptide repeat proteins that can cause neurodegeneration. We performed a genetic screen for regulators of RAN translation and identified small ribosomal protein subunit 25 (RPS25), presenting a potential therapeutic target for C9orf72-related amyotrophic lateral sclerosis and frontotemporal dementia and other neurodegenerative diseases caused by nucleotide repeat expansions.
Indexed as
AnimalsC9orf72 ProteinDNA Repeat ExpansionHumansNeurodegenerative DiseasesProtein BiosynthesisRibosomal ProteinsC9orf72 ProteinC9orf72 protein, humanRibosomal ProteinsRPS25 protein, human
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