Respiratory viral infections in otherwise healthy humans with inherited IRF7 deficiency

Autosomal recessive IRF7 deficiency was previously reported in three patients with single critical influenza or COVID-19 pneumonia episodes. The patients' fibroblasts and plasmacytoid dendritic cells produced no detectable type I and III IFNs, except IFN-β. Having discovered four new patients,...

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Veröffentlicht in:JOURNAL OF EXPERIMENTAL MEDICINE 2022-06, Vol.219 (7)
Hauptverfasser: Campbell, Tessa Mollie, Liu, Zhiyong, Zhang, Qian, Moncada-Velez, Marcela, Covill, Laura E, Zhang, Peng, Darazam, Ilad Alavi, Bastard, Paul, Bizien, Lucy, Bucciol, Giorgia, Enoksson, Sara Lind, Jouanguy, Emmanuelle, Karabela, Semsi Nur, Khan, Taushif, Kendir-Demirkol, Yasemin, Augusto Arias, Andres, Mansouri, Davood, Marits, Per, Marr, Nico, Migeotte, Isabelle, Moens, Leen, Ozcelik, Tayfun, Pellier, Isabelle, Sendel, Anton, Senoglu, Sevtap, Shahrooei, Mohammad, Smith, C.I. Edvard, Vandernoot, Isabelle, Willekens, Karen, Yasar, Kadriye Kart, Bergman, Peter, Abel, Laurent, Cobat, Aurelie, Casanova, Jean-Laurent, Meyts, Isabelle, Bryceson, Yenan T
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container_issue 7
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container_title JOURNAL OF EXPERIMENTAL MEDICINE
container_volume 219
creator Campbell, Tessa Mollie
Liu, Zhiyong
Zhang, Qian
Moncada-Velez, Marcela
Covill, Laura E
Zhang, Peng
Darazam, Ilad Alavi
Bastard, Paul
Bizien, Lucy
Bucciol, Giorgia
Enoksson, Sara Lind
Jouanguy, Emmanuelle
Karabela, Semsi Nur
Khan, Taushif
Kendir-Demirkol, Yasemin
Augusto Arias, Andres
Mansouri, Davood
Marits, Per
Marr, Nico
Migeotte, Isabelle
Moens, Leen
Ozcelik, Tayfun
Pellier, Isabelle
Sendel, Anton
Senoglu, Sevtap
Shahrooei, Mohammad
Smith, C.I. Edvard
Vandernoot, Isabelle
Willekens, Karen
Yasar, Kadriye Kart
Bergman, Peter
Abel, Laurent
Cobat, Aurelie
Casanova, Jean-Laurent
Meyts, Isabelle
Bryceson, Yenan T
description Autosomal recessive IRF7 deficiency was previously reported in three patients with single critical influenza or COVID-19 pneumonia episodes. The patients' fibroblasts and plasmacytoid dendritic cells produced no detectable type I and III IFNs, except IFN-β. Having discovered four new patients, we describe the genetic, immunological, and clinical features of seven IRF7-deficient patients from six families and five ancestries. Five were homozygous and two were compound heterozygous for IRF7 variants. Patients typically had one episode of pulmonary viral disease. Age at onset was surprisingly broad, from 6 mo to 50 yr (mean age 29 yr). The respiratory viruses implicated included SARS-CoV-2, influenza virus, respiratory syncytial virus, and adenovirus. Serological analyses indicated previous infections with many common viruses. Cellular analyses revealed strong antiviral immunity and expanded populations of influenza- and SARS-CoV-2-specific memory CD4+ and CD8+ T cells. IRF7-deficient individuals are prone to viral infections of the respiratory tract but are otherwise healthy, potentially due to residual IFN-β and compensatory adaptive immunity.
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Edvard ; Vandernoot, Isabelle ; Willekens, Karen ; Yasar, Kadriye Kart ; Bergman, Peter ; Abel, Laurent ; Cobat, Aurelie ; Casanova, Jean-Laurent ; Meyts, Isabelle ; Bryceson, Yenan T</creator><creatorcontrib>Campbell, Tessa Mollie ; Liu, Zhiyong ; Zhang, Qian ; Moncada-Velez, Marcela ; Covill, Laura E ; Zhang, Peng ; Darazam, Ilad Alavi ; Bastard, Paul ; Bizien, Lucy ; Bucciol, Giorgia ; Enoksson, Sara Lind ; Jouanguy, Emmanuelle ; Karabela, Semsi Nur ; Khan, Taushif ; Kendir-Demirkol, Yasemin ; Augusto Arias, Andres ; Mansouri, Davood ; Marits, Per ; Marr, Nico ; Migeotte, Isabelle ; Moens, Leen ; Ozcelik, Tayfun ; Pellier, Isabelle ; Sendel, Anton ; Senoglu, Sevtap ; Shahrooei, Mohammad ; Smith, C.I. 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title Respiratory viral infections in otherwise healthy humans with inherited IRF7 deficiency
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