ArticleCell reports. Medicine2023
Single-cell RNA-sequencing of PBMCs from SAVI patients reveals disease-associated monocytes with elevated integrated stress response.
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Article in Cell reports. Medicine, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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19citing papers in PubMed
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Who cites it
19 citing papers in PubMed, 12 citations in OpenAlex.
- The cGAS-STING/MITA pathway in innate antiviral immunity and beyond.Cell insight · 2026Review
- Atherosclerotic Cell Fates: A Single-Cell View of ER Stress.Journal of cardiovascular development and disease · 2026Review
- Single-Cell Analyses Reveal Dysregulation of Ribosomal Protein Genes During Hematopoietic Stem and Progenitor Cell Aging.Advanced biology · 2026Article
- Systematic dysregulation of immune-related alternative polyadenylation in systemic lupus erythematosus contributes to patient stratification.Journal of translational autoimmunity · 2026Article
- Lung-only involvement in STING-associated vasculopathy with onset in infancy: a diagnostic pitfall in the absence of cutaneous vasculitis.Zeitschrift fur Rheumatologie · 2026Article
- Interstitial lung disease and the STING pathway.The Journal of clinical investigation · 2026Review
- Identification of RPL4 as a biomarker associated with disulfidptosis and arachidonic acid metabolism in breast cancer.Discover oncology · 2026Article
- First successful allogeneic hematopoietic stem cell transplantation in STING-associated vasculopathy of infancy-A case report.Molecular therapy. Advances · 2026Article
- Pharmacological stabilization of hypoxia-inducible factor 1-α dampens the interferon response and promotes glycolysis in Aicardi-Goutières syndrome.Nature communications · 2026Article
- Overcoming immunotherapy resistance in breast cancer: a novel strategy by targeting the integrated stress response.Frontiers in cell and developmental biology · 2026Review
- SAVI: molecular mechanisms, clinical spectrum and precision medicine approaches beyond type-I IFN.Frontiers in immunology · 2026Review
- Single-cell multi-omic landscape reveals anatomical-specific immune features in adult and pediatric sepsis.Nature immunology · 2026Article
- Integrated single-cell analysis with experimental validation reveals ANXA2 as a therapeutic target for ferroptosis in inflammatory bowel disease.American journal of translational research · 2026Article
- Article
- Update on new autoinflammatory disorders from the 2024 Pediatric Rheumatology European Society Congress.Pediatric rheumatology online journal · 2025Review
- PRRs-Dependent and Independent Mechanisms of STING Signaling in Inflammatory and Autoimmune Diseases.Biomedicines · 2025Review
- [Research progress on the cGAS-STING signaling pathway in immune-mediated inflammatory diseases in children].Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2025Review
- Induction of the ISR by AB5 subtilase cytotoxin drives type-I IFN expression in pDCs via STING activation.Proceedings of the National Academy of Sciences of the United States of America · 2025Article
- Tissue inflammation induced by constitutively active STING is mediated by enhanced TNF signaling.eLife · 2025Article
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Authors and funding
34 authors at 8 institutions in 3 countries.
Camille de CevinsUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France; Sanofi R&D Data and Data Science, Artificial Intelligence & Deep Analytics, Omics Data Science, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Laure DelageUniversité de Paris Cité, Imagine Institute Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, INSERM UMR 1163, 75015 Paris, France; Checkpoint Immunology, Immunology and Inflammation Therapeutic Area, Sanofi, 94400 Vitry-sur-Seine, France.
Maxime BatignesUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France.
Quentin RillerUniversité de Paris Cité, Imagine Institute Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, INSERM UMR 1163, 75015 Paris, France.
Marine LukaUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France; Labtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, 75015 Paris, France.
Anne RemauryGenomics and Proteomics Groups, Translational Sciences, Sanofi R&D, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Boris SorinUniversité de Paris Cité, Imagine Institute Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, INSERM UMR 1163, 75015 Paris, France.
Tinhinane FaliUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France.
Cécile MassonBioinformatics Platform, Structure Fédérative de Recherche Necker, INSERM UMR1163, Université de Paris, Imagine Institute, Paris, France.
Bénédicte HoareauSorbonne Université, INSERM UMS037 PASS, Plateforme de Cytométrie (CyPS), Paris, France.
Catherine MeunierGenomics and Proteomics Groups, Translational Sciences, Sanofi R&D, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Mélanie ParisotGenomics Core Facility, Institut Imagine-Structure Fédérative de Recherche Necker, INSERM U1163 et INSERM US24/CNRS UAR3633, Paris Descartes Sorbonne Paris Cite University, Paris, France.
Mohammed ZarhrateGenomics Core Facility, Institut Imagine-Structure Fédérative de Recherche Necker, INSERM U1163 et INSERM US24/CNRS UAR3633, Paris Descartes Sorbonne Paris Cite University, Paris, France.
Brieuc P PérotUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France.
Víctor García-ParedesUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France.
Francesco CarboneUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France; Labtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, 75015 Paris, France.
Lou GalliotAix Marseille Université, CNRS, INSERM, CIML, 13288 Marseille Cedex 9, France.
Béatrice NalAix Marseille Université, CNRS, INSERM, CIML, 13288 Marseille Cedex 9, France.
Philippe PierreAix Marseille Université, CNRS, INSERM, CIML, 13288 Marseille Cedex 9, France; Institute of Biomedicine (iBiMED), Department of Medical Sciences, University of Aveiro, 3810-193 Aveiro, Portugal; Shanghai Institute of Immunology, Department of Microbiology and Immunology, Shanghai Jiao Tong University School of Medicine, Shanghai 200025, P.R. China.
Luc CanardGenomics and Proteomics Groups, Translational Sciences, Sanofi R&D, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Charlotte BoussardUniversité de Paris Cité, Imagine Institute Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, INSERM UMR 1163, 75015 Paris, France.
Etienne CrickxUniversité de Paris Cité, Imagine Institute Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, INSERM UMR 1163, 75015 Paris, France; Service de Médecine Interne, Centre national de référence des cytopénies auto-immunes de l'adulte, Hôpital Henri Mondor, Fédération Hospitalo-Universitaire TRUE InnovaTive theRapy for immUne disordErs, Assistance Publique Hôpitaux de Paris (AP-HP), Université Paris Est Créteil, Créteil, France.
Jean-Claude GuillemotGenomics and Proteomics Groups, Translational Sciences, Sanofi R&D, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Brigitte Bader-MeunierPediatric Immuno-hematology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP. Centre Université Paris Cité, 75015 Paris, France.
Alexandre BélotInternational Center of Infectiology Research (CIRI), University of Lyon, INSERM U1111, Claude Bernard University, Lyon 1, CNRS, UMR5308, ENS of Lyon, Lyon, France; National Reference Center for Rheumatic, Autoimmune and Systemic Diseases in Children (RAISE), Pediatric Nephrology, Rheumatology, Dermatology Unit, Hospital of Mother and Child, Hospices Civils of Lyon, Lyon, France.
Pierre QuartierPediatric Immuno-hematology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP. Centre Université Paris Cité, 75015 Paris, France.
Marie-Louise FrémondPediatric Immuno-hematology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP. Centre Université Paris Cité, 75015 Paris, France; Université Paris Cité, Imagine Institute, Laboratory of Neurogenetics and Neuroinflammation, INSERM UMR 1163, 75015 Paris, France.
Bénédicte NevenUniversité de Paris Cité, Imagine Institute Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, INSERM UMR 1163, 75015 Paris, France; Pediatric Immuno-hematology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP. Centre Université Paris Cité, 75015 Paris, France.
Galina BoldinaSanofi R&D Data and Data Science, Artificial Intelligence & Deep Analytics, Omics Data Science, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Franck AugéSanofi R&D Data and Data Science, Artificial Intelligence & Deep Analytics, Omics Data Science, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Fischer AlainUniversité de Paris, Imagine Institute, INSERM UMR 1163, 75015 Paris, France; Collège de France, Paris, France; Department of Paediatric Immuno-Haematology and Rheumatology, Reference Center for Rheumatic, AutoImmune and Systemic Diseases in Children (RAISE), Hôpital Necker-Enfants Malades, Assistance Publique - Hôpitaux de Paris (AP-HP) 75015 Paris, France.
Michel DidierGenomics and Proteomics Groups, Translational Sciences, Sanofi R&D, 1 Av Pierre Brossolette, 91385 Chilly-Mazarin, France.
Frédéric Rieux-LaucatUniversité de Paris Cité, Imagine Institute Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, INSERM UMR 1163, 75015 Paris, France.
Mickaël M MénagerUniversité de Paris Cité, Imagine Institute, Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, INSERM UMR 1163, 75015 Paris, France; Labtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, 75015 Paris, France. Electronic address: mickael.menager@institutimagine.org.
Inserm · FRSanofi (France) · FRInstitut des Maladies Génétiques Imagine · FRUniversité Paris Cité · FRAix-Marseille Université · FRCentre National de la Recherche Scientifique · FRAssistance Publique – Hôpitaux de Paris · FRSorbonne Université · FR
Funding
No grant is acknowledged in the PubMed record.
Abstract
Gain-of-function mutations in stimulator of interferon gene 1 (STING1) result in STING-associated vasculopathy with onset in infancy (SAVI), a severe autoinflammatory disease. Although elevated type I interferon (IFN) production is thought to be the leading cause of the symptoms observed in patients, STING can induce a set of pathways, which have roles in the onset and severity of SAVI and remain to be elucidated. To this end, we performed a multi-omics comparative analysis of peripheral blood mononuclear cells (PBMCs) and plasma from SAVI patients and healthy controls, combined with a dataset of healthy PBMCs treated with IFN-β. Our data reveal a subset of disease-associated monocyte, expressing elevated CCL3, CCL4, and IL-6, as well as a strong integrated stress response, which we suggest is the result of direct PERK activation by STING. Cell-to-cell communication inference indicates that these monocytes lead to T cell early activation, resulting in their senescence and apoptosis. Last, we propose a transcriptomic signature of STING activation, independent of type I IFN response.
Indexed as
Interferon Type IVascular DiseasesHumansLeukocytes, MononuclearMonocytesRNAInterferon Type IRNAintegrated stress responseSAVIscRNA-seqSTINGtype I interferonunfolded protein response
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