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Preexisting autoantibodies to type I IFNs underlie critical COVID-19 pneumonia in patients with APS-1.
Bastard, Paul; Orlova, Elizaveta; Sozaeva, Leila; Lévy, Romain; James, Alyssa; Schmitt, Monica M; Ochoa, Sebastian; Kareva, Maria; Rodina, Yulia; Gervais, Adrian; Le Voyer, Tom; Rosain, Jérémie; Philippot, Quentin; Neehus, Anna-Lena; Shaw, Elana; Migaud, Mélanie; Bizien, Lucy; Ekwall, Olov; Berg, Stefan; Beccuti, Guglielmo; Ghizzoni, Lucia; Thiriez, Gérard; Pavot, Arthur; Goujard, Cécile; Frémond, Marie-Louise; Carter, Edwin; Rothenbuhler, Anya; Linglart, Agnès; Mignot, Brigite; Comte, Aurélie; Cheikh, Nathalie; Hermine, Olivier; Breivik, Lars; Husebye, Eystein S; Humbert, Sébastien; Rohrlich, Pierre; Coaquette, Alain; Vuoto, Fanny; Faure, Karine; Mahlaoui, Nizar; Kotnik, Primoz; Battelino, Tadej; Trebusak Podkrajsek, Katarina; Kisand, Kai; Ferré, Elise M N; DiMaggio, Thomas; Rosen, Lindsey B; Burbelo, Peter D; McIntyre, Martin; Kann, Nelli Y.
Afiliación
  • Bastard P; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Orlova E; University of Paris, Imagine Institute, Paris, France.
  • Sozaeva L; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY.
  • Lévy R; Endocrinology Research Centre, Moscow, Russia.
  • James A; Endocrinology Research Centre, Moscow, Russia.
  • Schmitt MM; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Ochoa S; University of Paris, Imagine Institute, Paris, France.
  • Kareva M; Pediatric Immunology, Hematology and Rheumatology Unit, Necker Hospital for Sick Children, Assistance Publique Hôpitaux de Paris, Paris, France.
  • Rodina Y; Laboratory of Clinical Immunology and Microbiology, Division of Intramural Research, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
  • Gervais A; Laboratory of Clinical Immunology and Microbiology, Division of Intramural Research, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
  • Le Voyer T; Laboratory of Clinical Immunology and Microbiology, Division of Intramural Research, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
  • Rosain J; Endocrinology Research Centre, Moscow, Russia.
  • Philippot Q; Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology, Moscow, Russia.
  • Neehus AL; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Shaw E; University of Paris, Imagine Institute, Paris, France.
  • Migaud M; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Bizien L; University of Paris, Imagine Institute, Paris, France.
  • Ekwall O; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Berg S; University of Paris, Imagine Institute, Paris, France.
  • Beccuti G; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Ghizzoni L; University of Paris, Imagine Institute, Paris, France.
  • Thiriez G; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Pavot A; University of Paris, Imagine Institute, Paris, France.
  • Goujard C; Laboratory of Clinical Immunology and Microbiology, Division of Intramural Research, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
  • Frémond ML; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Carter E; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, Institut National de la Santé et de la Recherche Médicale U1163, Necker Hospital for Sick Children, Paris, France.
  • Rothenbuhler A; Department of Pediatrics, Institute of Clinical Sciences, The Sahlgrenska Academy, University of Gothenburg, Sweden.
  • Linglart A; Department of Rheumatology and Inflammation Research, Institute of Medicine, The Sahlgrenska Academy, University of Gothenburg, Sweden.
  • Mignot B; Department of Pediatrics, Institute of Clinical Sciences, The Sahlgrenska Academy, University of Gothenburg, Sweden.
  • Comte A; Department of Medical Sciences, University of Turin, Turin, Italy.
  • Cheikh N; Department of Medical Sciences, University of Turin, Turin, Italy.
  • Hermine O; Intensive Care Unit, Besançon Hospital, Besançon, France.
  • Breivik L; Intensive Care Unit, Kremlin-Bicêtre Hospital, Kremlin-Bicêtre, France.
  • Husebye ES; Internal Medicine Department, Bicêtre Hospital, Assistance Publique Hôpitaux de Paris, Paris Saclay University, Institut National de la Santé et de la Recherche Médicale U1018, Le Kremlin-Bicêtre, France.
  • Humbert S; Pediatric Immunology, Hematology and Rheumatology Unit, Necker Hospital for Sick Children, Assistance Publique Hôpitaux de Paris, Paris, France.
  • Rohrlich P; Laboratory of Neurogenetics and Neuroinflammation, Université de Paris, Imagine Institute, Paris, France.
  • Coaquette A; Centre for Genomic and Experimental Medicine, Medical Research Council Institute of Genetics and Molecular Medicine, Edinburgh, UK.
  • Vuoto F; Pediatric Endocrinology Department, Bicêtre Hospital, Assistance Publique Hôpitaux de Paris, Paris Saclay University, Le Kremlin-Bicêtre, France.
  • Faure K; Pediatric Endocrinology Department, Bicêtre Hospital, Assistance Publique Hôpitaux de Paris, Paris Saclay University, Le Kremlin-Bicêtre, France.
  • Mahlaoui N; Pediatric Medicine Unit, University Hospital of Besançon, Besançon, France.
  • Kotnik P; Pediatric Medicine Unit, University Hospital of Besançon, Besançon, France.
  • Battelino T; Pediatric Hematology Unit, University Hospital of Besançon, Besançon, France.
  • Trebusak Podkrajsek K; University of Paris, Imagine Institute, Paris, France.
  • Kisand K; Hematology department, University of Paris, Necker Hospital for Sick Children, Assistance Publique Hôpitaux de Paris, Paris, France.
  • Ferré EMN; Department of Clinical Science and K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Bergen, Norway.
  • DiMaggio T; Department of Clinical Science and K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Bergen, Norway.
  • Rosen LB; Department of Medicine, Haukeland University Hospital, Bergen, Norway.
  • Burbelo PD; Department of Medicine (Solna), Karolinska Institutet, Stockholm, Sweden.
  • McIntyre M; Internal Medicine Unit, Besançon Hospital, Besançon, France.
  • Kann NY; Pediatric Hematology and Oncology unit, Centre Hospitalier Universitaire de Nice, Nice, France.
J Exp Med ; 218(7)2021 07 05.
Article en En | MEDLINE | ID: mdl-33890986
ABSTRACT
Patients with biallelic loss-of-function variants of AIRE suffer from autoimmune polyendocrine syndrome type-1 (APS-1) and produce a broad range of autoantibodies (auto-Abs), including circulating auto-Abs neutralizing most type I interferons (IFNs). These auto-Abs were recently reported to account for at least 10% of cases of life-threatening COVID-19 pneumonia in the general population. We report 22 APS-1 patients from 21 kindreds in seven countries, aged between 8 and 48 yr and infected with SARS-CoV-2 since February 2020. The 21 patients tested had auto-Abs neutralizing IFN-α subtypes and/or IFN-ω; one had anti-IFN-ß and another anti-IFN-ε, but none had anti-IFN-κ. Strikingly, 19 patients (86%) were hospitalized for COVID-19 pneumonia, including 15 (68%) admitted to an intensive care unit, 11 (50%) who required mechanical ventilation, and four (18%) who died. Ambulatory disease in three patients (14%) was possibly accounted for by prior or early specific interventions. Preexisting auto-Abs neutralizing type I IFNs in APS-1 patients confer a very high risk of life-threatening COVID-19 pneumonia at any age.
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Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Neumonía / Autoanticuerpos / Interferón Tipo I / Poliendocrinopatías Autoinmunes / COVID-19 Límite: Adolescent / Adult / Child / Female / Humans / Male / Middle aged Idioma: En Año: 2021 Tipo del documento: Article
Texto completo
Imprimir
XML
PubMed Links
Buscar en Google

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Neumonía / Autoanticuerpos / Interferón Tipo I / Poliendocrinopatías Autoinmunes / COVID-19 Límite: Adolescent / Adult / Child / Female / Humans / Male / Middle aged Idioma: En Año: 2021 Tipo del documento: Article