Evidence map›Paper›PMID 32980424›Full record

ArticleThe Journal of allergy and clinical immunology2021

Coronavirus disease 2019 in patients with inborn errors of immunity: An international study.

Isabelle Meyts, Giorgia Bucciol, Isabella Quinti, Bénédicte Neven, Alain Fischer, Elena Seoane, Eduardo Lopez-Granados, Carla Gianelli, Angel Robles-Marhuenda, Pierre-Yves Jeandel and 46 more

Open access · bronzeAbstract readMulticenter Study
PubMed Full text at PMC Publisher
In one paragraph

Article in The Journal of allergy and clinical immunology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 219 papers, 6 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
219citing papers in PubMed, 6 pooled it
25.3field-weighted citation impact, top 1% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

219 citing papers in PubMed, 6 syntheses or guidelines pooled it, 360 citations in OpenAlex.

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159 more citing papers are in PubMed but not listed here.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

56 authors at 20 institutions in 13 countries.

Isabelle MeytsDepartment of Immunology and Microbiology, Inborn Errors of Immunity, Department of Pediatrics, University Hospitals Leuven and KU Leuven, Leuven, Belgium.
Giorgia BucciolDepartment of Immunology and Microbiology, Inborn Errors of Immunity, Department of Pediatrics, University Hospitals Leuven and KU Leuven, Leuven, Belgium.
Isabella QuintiDepartment of Molecular Medicine, Sapienza University of Rome, Rome, Italy.
Bénédicte NevenPediatric Hematology and Immunology Unit, Necker Hospital for Sick Children, Assistance Publique-Hopitaux de Paris, Paris, France; Université de Paris, Paris, France; Institut Imagine, Paris, France.
Alain FischerPediatric Hematology and Immunology Unit, Necker Hospital for Sick Children, Assistance Publique-Hopitaux de Paris, Paris, France; Université de Paris, Paris, France; Institut Imagine, Paris, France; Institut National de la Santé et de la Recherche Médicale (INSERM) UMR 1163, Paris, France; Collège de France, Paris, France.
Elena SeoaneDepartment of Pediatric Allergy and Immunology, and IISGM Gregorio Marañon University Hospital, Madrid, Spain.
Eduardo Lopez-GranadosUniversity Hospital La Paz and Lymphocyte Pathophysiology in Immunodeficiencies Group, IdiPAZ Institute for Health Research, Rare Disease Network Research Center (CIBERER), Madrid, Spain.
Carla GianelliUniversity Hospital La Paz and Lymphocyte Pathophysiology in Immunodeficiencies Group, IdiPAZ Institute for Health Research, Rare Disease Network Research Center (CIBERER), Madrid, Spain.
Angel Robles-MarhuendaUniversity Hospital La Paz and Lymphocyte Pathophysiology in Immunodeficiencies Group, IdiPAZ Institute for Health Research, Rare Disease Network Research Center (CIBERER), Madrid, Spain.
Pierre-Yves JeandelService de Médecine Interne, Centre Hospitalier Universitaire de Nice, Nice, France.
Catherine PaillardPediatric Oncohematology and Bone Marrow Transplantation Unit, Hôpital de Hautepierre, CHRU, Strasbourg, France.
Vijay G SankaranDivision of Hematology/Oncology, Boston Children's Hospital, Boston, Mass; Department of Pediatric Oncology, Dana-Farber Cancer Institute, Harvard Medical School, Boston, Mass.
Yesim Yilmaz DemirdagDivision of Basic and Clinical Immunology, Department of Medicine, University of California, Irvine, Calif.
Vassilios LougarisPediatrics Clinic and Institute for Molecular Medicine A. Nocivelli, Department of Clinical and Experimental Sciences, University of Brescia an ASST- Spedali Civili of Brescia, Brescia, Italy.
Alessandro AiutiSan Raffaele Telethon Institute for Gene Therapy (TIGET), Pediatric Immunohematology and Bone Marrow Transplantation Unit, IRCCS San Raffaele Scientific Institute Milan, Milan, Italy; Vita-Salute San Raffaele University, Milan, Italy.
Alessandro PlebaniPediatrics Clinic and Institute for Molecular Medicine A. Nocivelli, Department of Clinical and Experimental Sciences, University of Brescia an ASST- Spedali Civili of Brescia, Brescia, Italy.
Cinzia MilitoDepartment of Molecular Medicine, Sapienza University of Rome, Rome, Italy.
Virgil Ash DalmDepartment of Internal Medicine, Division of Clinical Immunology, Erasmus University Medical Center, Rotterdam, The Netherlands.
Kissy Guevara-HoyerDepartment of Immunology, IML and IdSSC, Hospital Clínico San Carlos, University Complutense of Madrid, Madrid, Spain.
Silvia Sánchez-RamónDepartment of Immunology, IML and IdSSC, Hospital Clínico San Carlos, University Complutense of Madrid, Madrid, Spain.
Liliana BezrodnikCenter for Clinical Immunology, Immunology Group Children's Hospital Ricardo Gutiérrez, Buenos Aires, Argentina.
Federica BarzaghiSan Raffaele Telethon Institute for Gene Therapy (TIGET), Pediatric Immunohematology and Bone Marrow Transplantation Unit, IRCCS San Raffaele Scientific Institute Milan, Milan, Italy.
Luis Ignacio Gonzalez-GranadoPrimary Immunodeficiencies Unit, Pediatrics, Hospital 12 Octubre, Madrid, Spain; Research Institute Hospital 12 Octubre (i+12), Madrid, Spain; Complutense University School of Medicine, Madrid, Spain.
Grant R HaymanImmunology Department, Epsom & St Helier University Hospitals NHS Trust, Carshalton, United Kingdom.
Gulbu UzelLaboratory of Clinical Immunology and Microbiology, National Institutes of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Leonardo Oliveira MendonçaDiscipline of Clinical Immunology and Allergy, Department of Internal Medicine, University of São Paulo School of Medicine, São Paulo, Brazil.
Carlo AgostiniDepartment of Medicine, Division of First Internal Medicine and Center for Immunologic Rare Disease, Ca' Foncello Treviso Hospital, University of Padua, Padua, Italy.
Giuseppe SpadaroDepartment of Translational Medical Sciences, Center for Basic and Clinical Immunology Research, University of Naples Federico II, Naples, Italy.
Raffaele BadolatoIstituto Molecolare "A Nocivelli," Department of Experimental and Clinical Sciences, University of Brescia & Asst Spedali civili, Brescia, Italy.
Annarosa SoresinaIstituto Molecolare "A Nocivelli," Department of Experimental and Clinical Sciences, University of Brescia & Asst Spedali civili, Brescia, Italy.
François VermeulenDepartment of Pediatrics, University Hospitals Leuven, Leuven, Belgium.
Cedric BosteelsLaboratory of Immunoregulation and Mucosal Immunology, VIB-UGent Center for Inflammation Research, Ghent, Belgium; Department of Internal Medicine and Pediatrics, Ghent University, Ghent, Belgium.
Bart N LambrechtLaboratory of Immunoregulation and Mucosal Immunology, VIB-UGent Center for Inflammation Research, Ghent, Belgium; Department of Internal Medicine and Pediatrics, Ghent University, Ghent, Belgium.
Michael KellerDivision of Allergy & Immunology, Children's National Hospital, Washington, DC.
Peter J MustilloDivision of Allergic Diseases and Immunology, Nationwide Children's Hospital, Columbus, Ohio.
Roshini S AbrahamDepartment of Pathology and Laboratory Medicine, Nationwide Children's Hospital, Columbus, Ohio.
Sudhir GuptaDivision of Basic and Clinical Immunology, Department of Medicine, University of California, Irvine, Calif.
Ahmet OzenDivision of Allergy and Immunology, Marmara University, Istanbul, Turkey; Isil Berat Barlan Center for Translational Medicine, Istanbul, Turkey; Istanbul Jeffrey Modell Diagnostic and Research Center for Primary Immunodeficiencies, Istanbul, Turkey.
Elif Karakoc-AydinerDivision of Allergy and Immunology, Marmara University, Istanbul, Turkey; Isil Berat Barlan Center for Translational Medicine, Istanbul, Turkey; Istanbul Jeffrey Modell Diagnostic and Research Center for Primary Immunodeficiencies, Istanbul, Turkey.
Safa BarisDivision of Allergy and Immunology, Marmara University, Istanbul, Turkey; Isil Berat Barlan Center for Translational Medicine, Istanbul, Turkey; Istanbul Jeffrey Modell Diagnostic and Research Center for Primary Immunodeficiencies, Istanbul, Turkey.
Alexandra F FreemanLaboratory of Clinical Immunology and Microbiology, National Institutes of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Marco Yamazaki-NakashimadaImmunodeficiencies Research Unit, National Institute of Pediatrics, Mexico City, Mexico.
Selma Scheffler-MendozaImmunodeficiencies Research Unit, National Institute of Pediatrics, Mexico City, Mexico.
Sara Espinosa-PadillaImmunodeficiencies Research Unit, National Institute of Pediatrics, Mexico City, Mexico.
Andrew R GenneryTranslational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom; Paediatric Immunology and Haematopoietic Stem Cell Transplantation, Great North Children's Hospital, Newcastle upon Tyne, United Kingdom.
Stephen JollesImmunodeficiency Centre for Wales, University Hospital of Wales, Cardiff, United Kingdom.
Yazmin EspinosaUniversidad del Desarrollo, Clínica Alemana de Santiago, Santiago de Chile, Chile; Hospital Roberto del Rio, Santiago, Chile.
M Cecilia PoliUniversidad del Desarrollo, Clínica Alemana de Santiago, Santiago de Chile, Chile; Hospital Roberto del Rio, Santiago, Chile.
Claire FieschiPediatric Hematology and Immunology Unit, Necker Hospital for Sick Children, Assistance Publique-Hopitaux de Paris, Paris, France; French National Reference Center for Primary Immune Deficiencies, Necker University, Paris, France; Department of Clinical Immunology, St-Louis Hospital-AP-HP, Paris, France.
Fabian HauckDepartment of Pediatrics, Dr von Hauner Children's Hospital, University Hospital, Ludwig-Maximilians-Universität München, Munich, Germany.
Charlotte Cunningham-RundlesDepartments of Medicine and Pediatrics, Mount Sinai School of Medicine, New York, NY.
Nizar MahlaouiPediatric Hematology and Immunology Unit, Necker Hospital for Sick Children, Assistance Publique-Hopitaux de Paris, Paris, France; French National Reference Center for Primary Immune Deficiencies, Necker University, Paris, France.
IUIS Committee of Inborn Errors of Immunity
Klaus WarnatzCenter for Chronic Immunodeficiency, University of Freiburg, Freiburg, Germany; Department of Rheumatology and Clinical Immunology, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany.
Kathleen E SullivanDivision of Allergy Immunology, Department of Pediatrics, The Children's Hospital of Philadelphia, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pa.
Stuart G TangyeGarvan Institute of Medical Research, Darlinghurst, Australia; St Vincent's Clinical School, UNSW Sydney, Darlinghurst, Australia. Electronic address: s.tangye@garvan.org.au.
University of Brescia · ITCentre for Biomedical Network Research on Rare Diseases · ESKU Leuven · BEMarmara University · TRHospital Clínico San Carlos · ESInstitut des Maladies Génétiques Imagine · FRNational Institutes of Health · USNationwide Children's Hospital · USSapienza University of Rome · ITUniversity of California, Irvine · USVIB-UGent Center for Inflammation Research · BEAssistance Publique – Hôpitaux de Paris · FRCentre Hospitalier Universitaire de Nice · FRChildren's National · USEpsom and St Helier University Hospitals NHS Trust · GBErasmus MC · NLGarvan Institute of Medical Research · AUHarvard University · USHôpital Saint-Louis · FRHôpitaux Universitaires de Strasbourg · FR

Funding

Regulation of IgA class switching in the intestinal mucosaP01AI061093 · NIAID · MOUNT SINAI SCHOOL OF MEDICINE OF NYU · PI CUNNINGHAM-RUNDLES, CHARLOTTE · 2004 to 2021
$29.8M
Primary Immune Deficiency ClinicZIAAI001247 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI FREEMAN, ALEXANDRA · 2019 to 2025
$8.7M
NIAID NIH HHS P01 AI061093
6 · The paper itself

Abstract

backgroundThere is uncertainty about the impact of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection in individuals with rare inborn errors of immunity (IEI), a population at risk of developing severe coronavirus disease 2019. This is relevant not only for these patients but also for the general population, because studies of IEIs can unveil key requirements for host defense.

objectiveWe sought to describe the presentation, manifestations, and outcome of SARS-CoV-2 infection in IEI to inform physicians and enhance understanding of host defense against SARS-CoV-2.

methodsAn invitation to participate in a retrospective study was distributed globally to scientific, medical, and patient societies involved in the care and advocacy for patients with IEI.

resultsWe gathered information on 94 patients with IEI with SARS-CoV-2 infection. Their median age was 25 to 34 years. Fifty-three patients (56%) suffered from primary antibody deficiency, 9 (9.6%) had immune dysregulation syndrome, 6 (6.4%) a phagocyte defect, 7 (7.4%) an autoinflammatory disorder, 14 (15%) a combined immunodeficiency, 3 (3%) an innate immune defect, and 2 (2%) bone marrow failure. Ten were asymptomatic, 25 were treated as outpatients, 28 required admission without intensive care or ventilation, 13 required noninvasive ventilation or oxygen administration, 18 were admitted to intensive care units, 12 required invasive ventilation, and 3 required extracorporeal membrane oxygenation. Nine patients (7 adults and 2 children) died.

conclusionsThis study demonstrates that (1) more than 30% of patients with IEI had mild coronavirus disease 2019 (COVID-19) and (2) risk factors predisposing to severe disease/mortality in the general population also seemed to affect patients with IEI, including more younger patients. Further studies will identify pathways that are associated with increased risk of severe disease and are nonredundant or redundant for protection against SARS-CoV-2.

Indexed as

SARS-CoV-2AdolescentAdultAgedChildChild, PreschoolCOVID-19FemaleGenetic Diseases, InbornHumansImmunologic Deficiency SyndromesInfantInfant, NewbornMaleMiddle AgedRetrospective StudiesCOVID-19hypogammaglobulinemiaimmune dysregulationinborn errors of immunityprimary immunodeficienciesSARS-CoV-2

Identifiers

PMID32980424
PMCPMC7832563
OpenAlexW3088841124

What OpenQuestion holds

Textmetadata
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Registered trials

None linked

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 OpenQuestion graph.