ArticleNature cell biology2025
A programmed decline in ribosome levels governs human early neurodevelopment.
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Article in Nature cell biology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.
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11citing papers in PubMed
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Who cites it
11 citing papers in PubMed.
- Illuminating ribosome biogenesis disorders through structural biology.RNA biology · 2026Review
- The Nucleolus in Human Disease: Ribosome Biogenesis, Nucleolar Surveillance, and Therapeutic Opportunities.Biomolecules · 2026Review
- Ribosomal RNA processing impairments in a B cell immunodeficient patient with WDR75 variants.Journal of human immunity · 2026Article
- Mathematical modeling of ribosome competition informs testable treatment strategies for drug-tolerant cancer persister cells.iScience · 2026Article
- RiboBright reveals cell-type-specific differences in ribosome organization and movement.Nature communications · 2026Article
- Signaling to make human ribosomes: Connections between the cytoplasm and the nucleolus.Molecular cell · 2026Review
- PP2Acα regulates cerebellar development via phosphorylation-dependent neuronal programs.iScience · 2026Article
- A single-cell transcriptomic atlas of the periventricular proliferative zone in the late gestation fetal brain in the pigtail macaque.Frontiers in neuroscience · 2026Article
- Systematic analysis of noncanonical ribosomal protein paralogs does not provide evidence for specialized functions inProceedings of the National Academy of Sciences of the United States of America · 2025Article
- Neurogenetic Disorders with Hearing Loss: Mechanisms, Classifications, and Emerging Insights.Current neurology and neuroscience reports · 2025Review
- High-dimensional co-expression network analysis reveals persistent TRH gene expression throughout axolotl telencephalon regeneration.Frontiers in bioinformatics · 2025Article
Corrections and comments
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Authors and funding
56 authors.
Chunyang Ni *Department of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Yudong Wei *Department of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Barbara Vona *Institute of Human Genetics, University Medical Center Göttingen, Göttingen, Germany.ORCID http://orcid.org/0000-0002-6719-3447
Dayea ParkDepartment of Molecular Biosciences, University of Texas at Austin, Austin, TX, USA.
Yulei WeiDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.ORCID http://orcid.org/0000-0002-4284-8558
Daniel A SchmitzDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Yi DingDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Masahiro SakuraiDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.ORCID http://orcid.org/0000-0002-6376-0299
Emily BallardDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Leijie LiDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA.ORCID http://orcid.org/0000-0001-9507-1183
Yan LiuMcDermott Center of Human Growth and Development, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Ashwani KumarMcDermott Center of Human Growth and Development, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Chao XingMcDermott Center of Human Growth and Development, University of Texas Southwestern Medical Center, Dallas, TX, USA.ORCID http://orcid.org/0000-0002-1838-0502
Shenlu QinDepartment of Physiology, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Sangin KimDepartment of Cancer Biology, Penn Center for Genome Integrity, Basser Center for BRCA, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.ORCID http://orcid.org/0000-0003-3961-6083
Martina FoglizzoAstbury Centre for Structural Molecular Biology, School of Molecular and Cellular Biology, Faculty of Biological Sciences, University of Leeds, Leeds, UK.ORCID http://orcid.org/0000-0001-9132-4737
Jianchao ZhaoState Key Laboratory of Organ Regeneration and Reconstruction, Institute of Zoology, Chinese Academy of Sciences, Beijing, China.
Hyung-Goo KimDepartment of Neurosurgery, Robert Wood Johnson Medical School, The State University of New Jersey, Rutgers, Piscataway, NJ, USA.
Cumhur EkmekciDNA Laboratories, Istanbul, Turkey.
Ehsan Ghayoor KarimianiCentre for Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, UK.
Shima ImannezhadDepartment of Pediatrics, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran.
Fatemeh EghbalDepartment of Medical Genetics, Next Generation Genetic Polyclinic, Mashhad, Iran.
Reza Shervin BadvChildren's Medical Center, Pediatrics Center of Excellence, Tehran University of Medical Sciences, Tehran, Iran.
Eva Maria Christina SchwaiboldInstitute of Human Genetics, Heidelberg University, Heidelberg, Germany.ORCID http://orcid.org/0000-0003-2708-9642
Mohammadreza DehghaniAbortion Research Centre, Yazd Reproductive Sciences Institute, Shahid Sadoughi University of Medical Sciences, Yazd, Iran.
Mohammad Yahya Vahidi MehrjardiDiabetes Research Center, Shahid Sadoughi University of Medical Sciences, Yazd, Iran.
Zahra MetanatGenetics of Non-Communicable Diseases Research Center, Zahedan University of Medical Sciences, Zahedan, Iran.
Hosein EslamiyehDepartment of Pediatrics, Shahid Sadoughi University of Medical Science, Yazd, Iran.
Ebtissal KhoujDepartment of Translational Genomics, Center for Genomic Medicine, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia.
Saleh Mohammed Nasser AlhajjMedical Genetics Department, King Faisal Hospital Specialist & Research Centre, Riyadh, Saudi Arabia.
Aziza ChedrawiDepartment of Neurosciences, King Faisal Hospital Specialist & Research Centre, Riyadh, Saudi Arabia.
Khushnooda RamzanDepartment of Clinical Genomics, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia.
Jamil A HashmiDepartment of Basic Medical Sciences, College of Medicine, Taibah University, Medina, Saudi Arabia.
Majed M AlluqmaniDepartment of Neurology, College of Medicine, Taibah University, Medina, Saudi Arabia.
Sulman BasitDepartment of Basic Medical Sciences, College of Medicine, Taibah University, Medina, Saudi Arabia.
Danai VeltraLaboratory of Medical Genetics, Medical School, National and Kapodistrian University of Athens, St. Sophia's Children's Hospital, Athens, Greece.ORCID http://orcid.org/0000-0002-6548-0928
Nikolaos M MarinakisLaboratory of Medical Genetics, Medical School, National and Kapodistrian University of Athens, St. Sophia's Children's Hospital, Athens, Greece.
Georgios NiotakisPediatric Neurology Department, Venizelion Hospital, Heraklion, Greece.
Pelagia VorgiaAgrifood and Life Sciences Institute, Hellenic Mediterranean University, Heraklion, Greece.
Christalena SofocleousLaboratory of Medical Genetics, Medical School, National and Kapodistrian University of Athens, St. Sophia's Children's Hospital, Athens, Greece.
Hane Lee3billion, Seoul, South Korea.
Won Chan Jeong3billion, Seoul, South Korea.
Muhammad UmairMedical Genomics Research Department, King Abdullah International Medical Research Center (KAIMRC), King Saud bin Abdulaziz University for Health Sciences, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia.
Muhammad BilalDepartment of Biotechnology, Begum Nusrat Bhutto Women University, Sukkur, Pakistan.
César Augusto Pinheiro Ferreira AlvesDepartment of Radiology, Division of Neuroradiology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Matthew SieberDepartment of Physiology, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Michael KruerPediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital, Phoenix, AZ, USA.
Henry HouldenDepartment of Neuromuscular Diseases, UCL Institute of Neurology, Queen Square, London, UK.ORCID http://orcid.org/0000-0002-2866-7777
Fowzan S AlkurayaDepartment of Translational Genomics, Center for Genomic Medicine, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia.ORCID http://orcid.org/0000-0003-4158-341X
Elton ZeqirajAstbury Centre for Structural Molecular Biology, School of Molecular and Cellular Biology, Faculty of Biological Sciences, University of Leeds, Leeds, UK.ORCID http://orcid.org/0000-0003-0239-5926
Roger A GreenbergDepartment of Cancer Biology, Penn Center for Genome Integrity, Basser Center for BRCA, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.ORCID http://orcid.org/0000-0003-1326-8981
Can CenikDepartment of Molecular Biosciences, University of Texas at Austin, Austin, TX, USA.ORCID http://orcid.org/0000-0001-6370-0889
Leqian YuDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA. leqianyu@ioz.ac.cn.ORCID http://orcid.org/0000-0003-2060-463X
Reza MaroofianDepartment of Neuromuscular Diseases, UCL Institute of Neurology, Queen Square, London, UK. r.maroofian@ucl.ac.uk.ORCID http://orcid.org/0000-0001-6763-1542
Jun WuDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA. Jun2.Wu@utsouthwestern.edu.ORCID http://orcid.org/0000-0001-9863-1668
Michael BuszczakDepartment of Molecular Biology, University of Texas Southwestern Medical Center, Dallas, TX, USA. Michael.Buszczak@utsouthwestern.edu.ORCID http://orcid.org/0000-0001-6361-3654
Funding
UT Southwestern Medical Center Simmons Comprehensive Cancer CenterP30CA142543 · NCI · UT SOUTHWESTERN MEDICAL CENTER · PI Kathryn Ann O'Donnell · 2010 to 2026
$53.7MDNA Double Strand Break Chromatin Alterations and Genome IntegrityR01GM101149 · NIGMS · UNIVERSITY OF PENNSYLVANIA · PI GREENBERG, ROGER A · 2013 to 2025
$4.0MGenomic analysis of the Multiplex, Autozygous Populations in Cerebral Palsy (MAP CP) cohort: a focused approach to a complex diseaseR01NS127108 · NINDS · UNIVERSITY OF ARIZONA · PI Michael C Kruer · 2023 to 2026
$2.6MInvestigating the role of maternal metabolic reprogramming in progeny physiology and agingR01AG067604 · NIA · UT SOUTHWESTERN MEDICAL CENTER · PI SIEBER, MATTHEW · 2020 to 2024
$2.2MGenetic Dissection of Germ Cell Differentiation and FunctionR35GM144043 · NIGMS · UT SOUTHWESTERN MEDICAL CENTER · PI Michael Buszczak · 2022 to 2026
$2.1MDissect the mechanisms underlying interspecies pluripotent stem cell competitionR01HD103627 · NICHD · UT SOUTHWESTERN MEDICAL CENTER · PI Michael Buszczak, Ondine B Cleaver · 2022 to 2026
$1.8MDissect formative pluripotency using cultured pluripotent stem cellsR01GM138565 · NIGMS · UT SOUTHWESTERN MEDICAL CENTER · PI WU, JUN · 2021 to 2025
$1.7MCharacterization of how mRNA translation influences reproductive agingR01AG079513 · NIA · UT SOUTHWESTERN MEDICAL CENTER · PI Michael Buszczak · 2022 to 2026
$1.7MStudy of mitochondria-depleted pluripotent stem cells in development and diseaseF31NS125906 · NINDS · UT SOUTHWESTERN MEDICAL CENTER · PI SCHMITZ, DANIEL ANDREW · 2022 to 2023
$70kCancer Prevention and Research Institute of Texas (Cancer Prevention Research Institute of Texas) RR180042Chinese Academy of Sciences (CAS) XDB0820000Deutsche Stiftung Friedensforschung (German Foundation for Peace Research) DFG VO 2138/7-1NCI NIH HHS P30 CA142543NIA NIH HHS R01 AG067604NIA NIH HHS R01 AG079513NICHD NIH HHS R01 HD103627NIGMS NIH HHS R01 GM101149NIGMS NIH HHS R01 GM138565NIGMS NIH HHS R35 GM144043NINDS NIH HHS F31 NS125906NINDS NIH HHS R01 NS127108RCUK | Biotechnology and Biological Sciences Research Council (BBSRC) BB/Z51522X/1U.S. Department of Health & Human Services | NIH | Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD) HD103627U.S. Department of Health & Human Services | NIH | National Cancer Institute (NCI) CA174905U.S. Department of Health & Human Services | NIH | National Institute of General Medical Sciences (NIGMS) GM101149U.S. Department of Health & Human Services | NIH | National Institute of General Medical Sciences (NIGMS) GM138565U.S. Department of Health & Human Services | NIH | National Institute of General Medical Sciences (NIGMS) R35GM144043U.S. Department of Health & Human Services | NIH | National Institute of Neurological Disorders and Stroke (NINDS) NS125906U.S. Department of Health & Human Services | NIH | National Institute on Aging (U.S. National Institute on Aging) R01AG067604U.S. Department of Health & Human Services | NIH | National Institute on Aging (U.S. National Institute on Aging) R01AG079513Welch Foundation F-2027-20230405Welch Foundation I-2088Wellcome TrustWellcome Trust (Wellcome) 222531/Z/21/Z
Abstract
Many neurodevelopmental defects are linked to genes involved in housekeeping functions, such as those encoding ribosome biogenesis factors. How reductions in ribosome biogenesis can result in tissue- and developmental-specific defects remains unclear. Here we describe variants in the ribosome biogenesis factor AIRIM/C1orf109 that are primarily associated with neurodevelopmental disorders. Using human cerebral organoids in combination with proteomic, single-cell RNA sequencing and single-organoid translation analyses, we identify a previously unappreciated drop in protein production during early brain development. We find that ribosome levels decrease during neuroepithelial differentiation, making differentiating cells particularly vulnerable to perturbations in ribosome biogenesis during this time. Reduced ribosome availability more profoundly impacts the translation of specific transcripts, disrupting both survival and cell fate commitment of transitioning neuroepithelia. Enhancing mTOR activity suppresses the growth and developmental defects associated with AIRIM/C1orf109 variants. This work provides evidence for the functional importance of regulated changes in global protein synthesis capacity during cellular differentiation.
Indexed as
BrainNeurodevelopmental DisordersNeurogenesisRibosomesTranscription FactorsCell DifferentiationGene Expression Regulation, DevelopmentalHumansOrganoidsProtein BiosynthesisProteomicsTOR Serine-Threonine KinasesMTOR protein, humanTOR Serine-Threonine KinasesTranscription Factors
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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 Socratic graph.