ArticleScience advances2024
Neddylation orchestrates the complex transcriptional and posttranscriptional program that drives Schwann cell myelination.
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Article in Science advances, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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6citing papers in PubMed
1.9field-weighted citation impact, top 16% of its field
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Who cites it
6 citing papers in PubMed, 7 citations in OpenAlex.
- Oligodendrocyte Egr2 Mediates the Beneficial Effect of Resocialization on the Social Ability of Socially Isolated Mice.Neuroscience bulletin · 2026Article
- Neddylation is indispensable for early meiotic progression in spermatocytes via destabilizing HORMAD1 by SCF ubiquitin E3 ligase during synapsis.Cell & bioscience · 2026Article
- MTSS1-dependent ubiquitin modifications mediated by FBXO44 remodel the actin cytoskeleton to promote gastric cancer progression.Molecular cancer · 2026Article
- Regulation of UBC12 expression and protein neddylation by PINK1 suggests a primate-specific function.Zoological research · 2025Article
- Neddylation regulates the development and function of glutamatergic neurons.Communications biology · 2025Article
- Lingo1 in the hippocampus contributes to cognitive dysfunction after anesthesia and surgery in aged mice.International journal of biological sciences · 2025Article
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Authors and funding
43 authors at 13 institutions in 4 countries.
Paula Ayuso-GarcíaGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-7737-9341
Alejandro Sánchez-RuedaGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0003-4477-4959
Sergio Velasco-AvilésGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-9672-2264
Miguel Tamayo-CaroCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0003-4536-3182
Aroa Ferrer-PinósGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0003-2799-9777
Cecilia Huarte-SebastianGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0001-8456-2497
Vanesa AlvarezGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0003-3410-3369
Cristina RiobelloGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-8854-6854
Selene Jiménez-VegaGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-5887-4523
Izaskun BuendiaGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-1021-7444
Jorge Cañas-MartinGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0001-9644-2935
Héctor Fernández-SusavilaGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0003-1972-9902
Adrián Aparicio-ReyGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0001-8313-0350
Eva M Esquinas-RománGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-6412-1405
Carlos Rodríguez PonteGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.
Romane GuhlGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0001-9212-3777
Nicolas LavilleGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0009-0001-7261-3696
Encarni Pérez-AndrésCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0003-1459-8966
José L LavínCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0003-0914-3211
Monika González-LopezCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0003-1506-0332
Nuria Macías CámaraCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0009-0004-0652-1832
Ana M AransayCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0002-8271-612X
Juan José LozanoCentro de Investigación Biomédica en Red de Enfermedades Hepáticas y Digestivas (CIBERehd), Instituto de Salud Carlos III, Madrid, Spain.ORCID 0000-0001-7613-3908
James D SutherlandCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0003-3229-793X
Rosa BarrioCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0002-9663-0669
María Luz Martinez-ChantarCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0003-2765-6432
Mikel AzkargortaCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.
Félix ElortzaCenter for Cooperative Research in Biosciences (CIC bioGUNE), Basque Research and Technology Alliance (BRTA), Bizkaia Technology Park, 48160 Derio, Spain.ORCID 0000-0001-8839-5438
Mario Soriano-NavarroElectron Microscopy Core Facility, Centro de Investigación Príncipe Felipe (CIPF), 46012 Valencia, Spain.ORCID 0000-0002-7245-2095
Carlos MatuteLaboratory of Neurobiology, Achucarro Basque Center for Neuroscience, Science Park of UPV/EHU, Sede building, 48940 Leioa, Spain.ORCID 0000-0001-8672-711X
María Victoria Sánchez-GómezLaboratory of Neurobiology, Achucarro Basque Center for Neuroscience, Science Park of UPV/EHU, Sede building, 48940 Leioa, Spain.ORCID 0000-0001-5730-8194
Laura Bayón-CorderoLaboratory of Neurobiology, Achucarro Basque Center for Neuroscience, Science Park of UPV/EHU, Sede building, 48940 Leioa, Spain.ORCID 0000-0001-5551-3290
Alberto Pérez-SamartínLaboratory of Neurobiology, Achucarro Basque Center for Neuroscience, Science Park of UPV/EHU, Sede building, 48940 Leioa, Spain.ORCID 0000-0003-0834-0424
Susana B BravoProteomic Unit, Health Research Institute of Santiago de Compostela (IDIS), 15705 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0001-7615-3079
Thimo KurzEvotec SE, Innovation Dr, Milton, Abingdon OX14 4RT, UK and School of Molecular Biosciences, University of Glasgow, Glasgow G12 8QQ, UK.ORCID 0000-0003-0700-6993
Tomas Lama-DíazDNA Repair and Genome Integrity Laboratory, CIMUS, University of Santiago de Compostela-Instituto de Investigación Sanitaria, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-8568-2375
Miguel G BlancoDNA Repair and Genome Integrity Laboratory, CIMUS, University of Santiago de Compostela-Instituto de Investigación Sanitaria, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-2883-7326
Saif HaddadCentre for Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, UK.ORCID 0009-0008-9935-0081
Christopher J RecordCentre for Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, UK.ORCID 0000-0002-9802-2683
Peter M van HasseltDepartment of Metabolic Diseases, Division Pediatrics, Wilhelmina Children's Hospital University Medical Center Utrecht, Utrecht University, 3584 EA, Utrecht, Netherlands.ORCID 0000-0001-9821-8398
Mary M ReillyCentre for Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, UK.ORCID 0000-0003-0686-905X
Marta Varela-ReyGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-8395-2837
Ashwin WoodhooGene Regulatory Control in Disease Laboratory, Center for Research in Molecular Medicine and Chronic Diseases (CIMUS), Instituto de Investigación Sanitaria de Santiago de Compostela (IDIS), University of Santiago de Compostela, 15706 Santiago de Compostela, A Coruña, Spain.ORCID 0000-0002-8395-2837
Instituto de Investigación Sanitaria de Santiago · ESCIC bioGUNE · ESUniversity of the Basque Country · ESInstituto de Salud Carlos III · ESNational Hospital for Neurology and Neurosurgery · GBUniversidade de Santiago de Compostela · ESUniversité Paris Cité · FRAchucarro Basque Center for Neuroscience · ESCentro de Investigacion Principe Felipe · ESEuskadiko Parke Teknologikoa · ESEvotec (United Kingdom) · GBIkerbasque · ESWilhelmina Children's Hospital · NL
Funding
Wellcome Trust
Abstract
Myelination is essential for neuronal function and health. In peripheral nerves, >100 causative mutations have been identified that cause Charcot-Marie-Tooth disease, a disorder that can affect myelin sheaths. Among these, a number of mutations are related to essential targets of the posttranslational modification neddylation, although how these lead to myelin defects is unclear. Here, we demonstrate that inhibiting neddylation leads to a notable absence of peripheral myelin and axonal loss both in developing and regenerating mouse nerves. Our data indicate that neddylation exerts a global influence on the complex transcriptional and posttranscriptional program by simultaneously regulating the expression and function of multiple essential myelination signals, including the master transcription factor EGR2 and the negative regulators c-Jun and Sox2, and inducing global secondary changes in downstream pathways, including the mTOR and YAP/TAZ signaling pathways. This places neddylation as a critical regulator of myelination and delineates the potential pathogenic mechanisms involved in CMT mutations related to neddylation.
Indexed as
Charcot-Marie-Tooth DiseaseSchwann CellsAnimalsMiceMutationMyelin SheathProtein Processing, Post-Translational
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