ArticleNature neuroscience2015
Mutations in NONO lead to syndromic intellectual disability and inhibitory synaptic defects.
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Article in Nature neuroscience, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 62 papers, 1 of them a synthesis that pooled it.
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62citing papers in PubMed, 1 pooled it
3.9field-weighted citation impact, top 6% of its field
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Who cites it
62 citing papers in PubMed, 1 synthesis or guideline pooled it, 94 citations in OpenAlex.
- Genetic aetiology of global developmental delay and intellectual disability in Africa: a scoping review.Frontiers in genetics · 2026Pooled it
- Engineering a protein homodimer from a heterodimer: A chimeric DBHS protein.Protein science : a publication of the Protein Society · 2026Article
- Sex-specific metabolic regulation by the Drosophila RNA-binding protein Nab2.bioRxiv : the preprint server for biology · 2026Article
- Heart Transplant for Noncompaction Cardiomyopathy in NONO-Related Syndromic Intellectual Disability.Molecular genetics & genomic medicine · 2026Article
- NONO-Related Syndromic X-Linked Developmental Disability 34: Further Clinical and Molecular Delineation in a Prenatal Cohort.Prenatal diagnosis · 2026Article
- Case Report: A female case of X-linked intellectual disability syndrome type 34 caused by a NONO frameshift variant and literature review.Frontiers in pediatrics · 2026Article
- Chromatin Meets Condensates: Emerging Interplays Linking Nuclear Paraspeckles to Gene Activation.Epigenetics reports · 2026Article
- Multi-Omics Analysis Reveals Nono-Kcnq2 Regulation of Neuronal Excitability in Chronic Constriction Injury-Induced Neuropathic Pain.Research (Washington, D.C.) · 2026Article
- The gene-regulating proteins NONO and SFPQ assemble into ordered filaments.Communications biology · 2025Article
- Unraveling and controlling late-onset neurotoxicity of antisense oligonucleotides through strategic chemical modifications.Molecular therapy. Nucleic acids · 2025Article
- Muscle Cathepsin B Treatment Improves Behavioral and Neurogenic Deficits in a Mouse Model of Alzheimer's Disease.Aging cell · 2025Article
- Comparative Single-Cell Transcriptome Analysis of c-Met Receptor Expressing and Non-Expressing Projection Neurons in the Developing Frontal and Visual Cortices.Developmental neuroscience · 2025Article
- Paraspeckle protein NONO regulates active chromatin by allosterically stimulating NSD1.Cell reports · 2025Article
- Stress-Induced Membraneless Organelles in Neurons: Bridging Liquid-Liquid Phase Separation and Neurodevelopmental Dysfunction.International journal of molecular sciences · 2025Review
- Pigmented perivascular epithelioid cell tumors of the orbit withAmerican journal of ophthalmology case reports · 2025Article
- The NONO protein regulates nonclassical DNA structure: Effects on circadian genes and DNA damage.iScience · 2025Article
- PROSER1 modulates DNA demethylation through dual mechanisms to prevent syndromic developmental malformations.Genes & development · 2024Article
- Swine NONO promotes IRF3-mediated antiviral immune response by Detecting PRRSV N protein.PLoS pathogens · 2024Article
- An N-terminal and ankyrin repeat domain interactome of Shank3 identifies the protein complex with the splicing regulator Nono in mice.Genes to cells : devoted to molecular & cellular mechanisms · 2024Article
- X-linked intellectual developmental disorder with onset of neonatal heart failure: A case report and literature review.Molecular genetics and metabolism reports · 2024Article
2 more citing papers are in PubMed but not listed here.
Corrections and comments
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Authors and funding
30 authors at 4 institutions in 4 countries.
Dennis MircsofChronobiology and Sleep Research Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.
Maéva LangouëtINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Marlène RioINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Sébastien MouttonINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.ORCID http://orcid.org/0000-0002-8942-6731
Karine Siquier-PernetINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Christine Bole-FeysotGenomic Platform, INSERM UMR 1163, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Nicolas CagnardBioinformatic Platform, INSERM UMR 1163, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Patrick NitschkeBioinformatic Platform, INSERM UMR 1163, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Ludmila GasparChronobiology and Sleep Research Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.
Matej ŽnidaričChronobiology and Sleep Research Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.
Olivier AlibeuGenomic Platform, INSERM UMR 1163, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Ann-Kristina FritzInstitute of Anatomy, University of Zürich and Institute of Human Movement Sciences and Sport, ETH Zürich, Switzerland.
David P WolferInstitute of Anatomy, University of Zürich and Institute of Human Movement Sciences and Sport, ETH Zürich, Switzerland.
Aileen SchröterMolecular Imaging and Functional Pharmacology Group, University of Zürich, Zürich, Switzerland.
Giovanna BosshardNeuromorphology Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.
Markus RudinMolecular Imaging and Functional Pharmacology Group, University of Zürich, Zürich, Switzerland.
Christina KoesterNeuromorphology Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.
Florence CrestaniNeuromorphology Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.
Petra SeebeckCenter for Integrative Rodent Physiology, University of Zürich, Zürich, Switzerland.
Nathalie BoddaertINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Katrina PrescottYorkshire Regional Genetics Service, Leeds Teaching Hospitals National Health Service Trust, Department of Clinical Genetics, Chapel Allerton Hospital, Chapeltown Road, Leeds, UK.
DDD Study
Rochelle HinesTufts University, Sackler School of Graduate Biomedical Sciences, Boston, Massachusetts, USA.
Steven J MossTufts University, Sackler School of Graduate Biomedical Sciences, Boston, Massachusetts, USA.
Jean-Marc FritschyNeuromorphology Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.ORCID http://orcid.org/0000-0002-8552-3702
Arnold MunnichINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Jeanne AmielINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Steven A BrownChronobiology and Sleep Research Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.
Shiva K TyagarajanNeuromorphology Group, Institute of Pharmacology and Toxicology, University of Zürich, Zürich, Switzerland.ORCID http://orcid.org/0000-0003-0074-1805
Laurence ColleauxINSERM UMR 1163, Laboratory of Molecular and Pathophysiological Bases of Cognitive Disorders, Paris Descartes-Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Hospital, Paris, France.
Délégation Paris 5 · FRUniversity of Zurich · CHTufts University · USInstitut de France · FR
Funding
Mechanisms that determine Glutamine synthetase activity in the brainR01NS081986 · NINDS · TUFTS UNIVERSITY BOSTON · PI MOSS, STEPHEN J · 2012 to 2022
$3.7MConstructing inhibitory synapsesR01NS051195 · NINDS · UNIVERSITY OF PENNSYLVANIA · PI MOSS, STEPHEN J · 2006 to 2014
$3.2MModulation of GABAB receptor signalingR01NS048045 · NINDS · UNIVERSITY OF PENNSYLVANIA · PI MOSS, STEPHEN J · 2005 to 2010
$2.7MDissecting Mechanisms of GABAB-GIRK Plasticity with PsychostimulantsR01DA037170 · NIDA · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI MOSS, STEPHEN J, SLESINGER, PAUL A · 2014 to 2018
$2.1MNovel actions of neurosteroids on GABA (A) receptor traffickingR01MH097446 · NIMH · TUFTS UNIVERSITY BOSTON · PI DAVIES, PAUL ANDREW, MOSS, STEPHEN J · 2013 to 2017
$2.1MDeficits in KCC2 activity and the pathophysiology of Status EpilepticusR01NS087662 · NINDS · TUFTS UNIVERSITY BOSTON · PI MOSS, STEPHEN J · 2014 to 2018
$2.0MThe role of ubiquitination in regulating GABA(A) receptor functional expressionR01NS056359 · NINDS · UNIVERSITY OF PENNSYLVANIA · PI MOSS, STEPHEN J · 2007 to 2010
$1.4MDeficits in KCC2 activity and the pathophysiology of Autism spectrum disordersR21MH106954 · NIMH · TUFTS UNIVERSITY BOSTON · PI MOSS, STEPHEN J · 2015 to 2016
$454kImpaired KCC2 function underlies pharmacoresistant seizuresR21NS080064 · NINDS · TUFTS UNIVERSITY BOSTON · PI MOSS, STEPHEN J · 2013 to 2014
$452kDepartment of HealthNIDA NIH HHS R01 DA037170NIMH NIH HHS R01 MH097446NIMH NIH HHS R21 MH106954NINDS NIH HHS R01 NS048045NINDS NIH HHS R01 NS051195NINDS NIH HHS R01 NS056359NINDS NIH HHS R01 NS081986NINDS NIH HHS R01 NS087662NINDS NIH HHS R21 NS080064Wellcome TrustWellcome Trust WT098051
Abstract
The NONO protein has been characterized as an important transcriptional regulator in diverse cellular contexts. Here we show that loss of NONO function is a likely cause of human intellectual disability and that NONO-deficient mice have cognitive and affective deficits. Correspondingly, we find specific defects at inhibitory synapses, where NONO regulates synaptic transcription and gephyrin scaffold structure. Our data identify NONO as a possible neurodevelopmental disease gene and highlight the key role of the DBHS protein family in functional organization of GABAergic synapses.
Indexed as
AdolescentAnimalsBrainCells, CulturedDNA-Binding ProteinsHumansIntellectual DisabilityMaleMiceMice, Inbred C57BLMice, TransgenicMutationNeural InhibitionNuclear Matrix-Associated ProteinsOctamer Transcription FactorsPedigreeDNA-Binding ProteinsNONO protein, humanNuclear Matrix-Associated ProteinsOctamer Transcription FactorsRNA-Binding Proteins
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