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

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dc.contributor.authorMeyts, Isabelle
dc.contributor.authorBucciol, Giorgia
dc.contributor.authorQuinti, Isabella
dc.contributor.authorNeven, Bénédicte
dc.contributor.authorFischer, Alain
dc.contributor.authorSeoane, Elena
dc.contributor.authorLopez-Granados, Eduardo
dc.contributor.authorGianelli, Carla
dc.contributor.authorRobles-Marhuenda, Angel
dc.contributor.authorJeandel, Pierre-Yves
dc.contributor.authorPaillard, Catherine
dc.contributor.authorSankaran, Vijay G.
dc.contributor.authorDemirdag, Yesim Yilmaz
dc.contributor.authorLougaris, Vassilios
dc.contributor.authorAiuti, Alessandro
dc.contributor.authorPlebani, Alessandro
dc.contributor.authorMilito, Cinzia
dc.contributor.authorDalm, Virgil Ash
dc.contributor.authorGuevara-Hoyer, Kissy
dc.contributor.authorSánchez-Ramón, Silvia
dc.contributor.authorBezrodnik, Liliana
dc.contributor.authorBarzaghi, Federica
dc.contributor.authorGonzález-Granado, Luis Ignacio
dc.contributor.authorHayman, Grant R.
dc.contributor.authorUzel, Gulbu
dc.contributor.authorMendonça, Leonardo Oliveira
dc.contributor.authorAgostini, Carlo
dc.contributor.authorSpadaro, Giuseppe
dc.contributor.authorBadolato, Raffaele
dc.contributor.authorSoresina, Annarosa
dc.contributor.authorVermeulen, Francois
dc.contributor.authorBosteels, Cedric
dc.contributor.authorLambrecht, Bart N.
dc.contributor.authorKeller, Michael
dc.contributor.authorMustillo, Peter J.
dc.contributor.authorAbraham, Roshini S.
dc.contributor.authorGupta, Sudhir
dc.contributor.authorOzen, Ahmet
dc.contributor.authorKarakoc-Aydiner, Elif
dc.contributor.authorBaris, Safa
dc.contributor.authorFreeman, Alexandra F.
dc.contributor.authorYamazaki-Nakashimada, Marco
dc.contributor.authorScheffler-Mendoza, Selma
dc.contributor.authorEspinoza Padilla, Sara
dc.contributor.authorGennery, Andrew R.
dc.contributor.authorJolles, Stephen
dc.contributor.authorEspinosa, Yazmin
dc.contributor.authorPoli, Cecilia
dc.contributor.authorFieschi, Claire
dc.contributor.authorHauck, Fabian
dc.contributor.authorCunningham-Rundles, Charlotte
dc.contributor.authorMahlaouie, Nizar
dc.contributor.authorSullivan, Kathleen E.
dc.contributor.authorTangye, Stuart G.
dc.contributor.authorIUIS Committee of Inborn Errors of Immunity;
dc.date.accessioned2022-04-28T17:36:08Z
dc.date.available2022-04-28T17:36:08Z
dc.date.issued2022
dc.description.abstractBackground: There 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. Objective: We 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. Methods: An 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. Results: We 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. Conclusions: This 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.es
dc.description.versionVersión Publicadaes
dc.identifier.citationMeyts I, Bucciol G, Quinti I, Neven B, Fischer A, Seoane E, Lopez-Granados E, Gianelli C, Robles-Marhuenda A, Jeandel PY, Paillard C, Sankaran VG, Demirdag YY, Lougaris V, Aiuti A, Plebani A, Milito C, Dalm VA, Guevara-Hoyer K, Sánchez-Ramón S, Bezrodnik L, Barzaghi F, Gonzalez-Granado LI, Hayman GR, Uzel G, Mendonça LO, Agostini C, Spadaro G, Badolato R, Soresina A, Vermeulen F, Bosteels C, Lambrecht BN, Keller M, Mustillo PJ, Abraham RS, Gupta S, Ozen A, Karakoc-Aydiner E, Baris S, Freeman AF, Yamazaki-Nakashimada M, Scheffler-Mendoza S, Espinosa-Padilla S, Gennery AR, Jolles S, Espinosa Y, Poli MC, Fieschi C, Hauck F, Cunningham-Rundles C, Mahlaoui N; IUIS Committee of Inborn Errors of Immunity, Warnatz K, Sullivan KE, Tangye SG. Coronavirus disease 2019 in patients with inborn errors of immunity: An international study. J Allergy Clin Immunol. 2021 Feb;147(2):520-531. doi: 10.1016/j.jaci.2020.09.010.es
dc.identifier.urihttps://doi.org/10.1016/j.jaci.2020.09.010es
dc.identifier.urihttp://hdl.handle.net/11447/6032
dc.language.isoenes
dc.subjectSars-CoV-2es
dc.subjectCovid-19es
dc.subjectPrimary immunodeficiencieses
dc.subjectInborn errors of immunityes
dc.subjectHypogammaglobulinemiaes
dc.subjectImmune dysregulationes
dc.titleCoronavirus disease 2019 in patients with inborn errors of immunity: An international studyes
dc.typeArticlees
dcterms.sourceJournal of Allergy and Clinical Immunologyes

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