ArticleScience (New York, N.Y.)2021
tRNA overexpression rescues peripheral neuropathy caused by mutations in tRNA synthetase.
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Article in Science (New York, N.Y.), 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 86 papers.
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86citing papers in PubMed
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Who cites it
86 citing papers in PubMed.
- White matter disorders at the intersection of transcription, RNA processing and translation.Nature reviews. Neurology · 2026Review
- Therapeutic rescue of pathogenic asparaginyl-tRNA synthetase alleles.Molecular therapy. Nucleic acids · 2026Article
- The neuropathy-causingbioRxiv : the preprint server for biology · 2026Article
- Human aminoacyl-tRNA synthetases as integrators of translation and cell signalling networks.Nature reviews. Molecular cell biology · 2026Review
- RNA Polymerase III subunit Polr3a is required for craniofacial cartilage and bone development in zebrafish.PLoS genetics · 2026Article
- Locally synthesized glycyl aminoacyl-tRNA synthetase is important for local translation in neurons.Life science alliance · 2026Article
- Tuning tRNA synthetase inhibition reveals parabolic induction of stress granules limited in size and RNA content.RNA (New York, N.Y.) · 2026Article
- Aminoacyl-tRNA Synthetases: Variant Classification, Functional Assays, and Emerging Therapeutic Strategies.Journal of inherited metabolic disease · 2026Review
- A class of deep intronicmedRxiv : the preprint server for health sciences · 2026Article
- The regulation, function and disease relevance of cytoplasmic tRNAs.Nature reviews. Molecular cell biology · 2026Review
- tRNA synthetase activity is required for stress granule and P-body assembly.Genes & development · 2026Article
- Replating Induces mTOR-Dependent Rescue of Protein Synthesis in Charcot-Marie-Tooth Diseased Neurons.eNeuro · 2026Article
- A disease-causing isoleucyl-tRNA synthetase variant leads to altered protein complex formation and cellular stress response.The Journal of biological chemistry · 2026Article
- GTPBP2 inactivates Hippo signaling to promote triple-negative breast cancer cell malignancy.Scientific reports · 2026Article
- Mistranslation from an endogenous tRNA variant in human pan-genome cell lines.Nucleic acids research · 2026Article
- Targeting tRNA-Arg-TCT-4-1 suppresses cancer cell growth and tumorigenesis.bioRxiv : the preprint server for biology · 2026Article
- Axonal Eif5a hypusination controls local translation and mitigates defects in FUS-ALS.Nature neuroscience · 2026Article
- Inhibition of the integrated stress response rescues a histidyl-tRNA synthetase variant associated with Charcot-Marie-Tooth disease.NAR molecular medicine · 2026Article
- Modelling Neural Disorders with theBiomolecules · 2025Review
- Biallelic Variants in the DARS2 Gene as a Novel Cause of Axonal Charcot-Marie-Tooth Disease.Annals of neurology · 2025Article
26 more citing papers are in PubMed but not listed here.
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Authors and funding
17 authors.
Amila Zuko *Department of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0001-5423-4273
Moushami Mallik *Department of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0002-0970-7411
Robin ThompsonBiochemistry and Molecular Biology, Department of Chemistry, University of Hamburg, Hamburg, Germany.ORCID 0000-0003-4749-7707
Emily L SpauldingThe Jackson Laboratory, Bar Harbor, ME, USA.ORCID 0000-0001-9680-6348
Anne R WienandDepartment of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0001-6711-4713
Marije BeenDepartment of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0003-3856-4605
Abigail L D TadenevThe Jackson Laboratory, Bar Harbor, ME, USA.ORCID 0000-0003-0801-8267
Nick van BakelDepartment of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0002-3902-9914
Céline SijlmansDepartment of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.
Leonardo A SantosBiochemistry and Molecular Biology, Department of Chemistry, University of Hamburg, Hamburg, Germany.ORCID 0000-0001-5650-8098
Julia BussmannMolecular Neurogenetics Laboratory, Max Planck Institute for Molecular Biomedicine, Münster, Germany.
Marica CatinozziDepartment of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0002-9211-9951
Sarada DasBiochemistry and Molecular Biology, Department of Chemistry, University of Hamburg, Hamburg, Germany.ORCID 0000-0002-6595-533X
Divita KulshresthaDepartment of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0002-6188-6795
Robert W BurgessThe Jackson Laboratory, Bar Harbor, ME, USA.ORCID 0000-0002-9229-3407
Zoya IgnatovaBiochemistry and Molecular Biology, Department of Chemistry, University of Hamburg, Hamburg, Germany.ORCID 0000-0002-9478-8825
Erik StorkebaumDepartment of Molecular Neurobiology, Donders Institute for Brain, Cognition and Behaviour and Faculty of Science, Radboud University, Nijmegen, Netherlands.ORCID 0000-0002-9519-5642
Funding
The Jackson Laboratory Center for Precision Genetics: From New Models to Novel TherapeuticsU54OD020351 · OD · JACKSON LABORATORY · PI LUTZ, CATHLEEN M · 2015 to 2019
$10.2MGenetics of the Neuromuscular Junction: Mechanisms and Disease ModelsR01NS054154 · NINDS · JACKSON LABORATORY · PI BURGESS, ROBERT W. · 2006 to 2019
$6.0MThe Genetics of the Neuromuscular Junction: Mechanisms and Disease ModelsR37NS054154 · NINDS · JACKSON LABORATORY · PI Robert W. Burgess · 2020 to 2026
$5.9MExploring the Functions of tRNA Synthetases in the Nucleus and their Relationship to CMTR01NS113583 · NINDS · SCRIPPS RESEARCH INSTITUTE, THE · PI BURGESS, ROBERT W., YANG, XIANG-LEI · 2020 to 2024
$2.9MNIH HHS U54 OD020351NINDS NIH HHS R01 NS054154NINDS NIH HHS R01 NS113583NINDS NIH HHS R37 NS054154
Abstract
Heterozygous mutations in six transfer RNA (tRNA) synthetase genes cause Charcot-Marie-Tooth (CMT) peripheral neuropathy. CMT mutant tRNA synthetases inhibit protein synthesis by an unknown mechanism. We found that CMT mutant glycyl-tRNA synthetases bound tRNA
Indexed as
AnimalsCharcot-Marie-Tooth DiseaseDisease Models, AnimalDrosophila melanogasterFemaleGlycine-tRNA LigaseHumansMaleMiceMice, TransgenicMotor NeuronsRNA, Transfer, GlyGlycine-tRNA LigaseRNA, Transfer, Gly
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.