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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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. Edvard ; Vandernoot, Isabelle ; Willekens, Karen ; Yasar, Kadriye Kart ; Bergman, Peter ; Abel, Laurent ; Cobat, Aurelie ; Casanova, Jean-Laurent ; Meyts, Isabelle ; Bryceson, Yenan T</creatorcontrib><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.</description><identifier>ISSN: 0022-1007</identifier><language>eng</language><publisher>ROCKEFELLER UNIV PRESS</publisher><ispartof>JOURNAL OF EXPERIMENTAL MEDICINE, 2022-06, Vol.219 (7)</ispartof><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,315,777,781,27841</link.rule.ids></links><search><creatorcontrib>Campbell, Tessa Mollie</creatorcontrib><creatorcontrib>Liu, Zhiyong</creatorcontrib><creatorcontrib>Zhang, Qian</creatorcontrib><creatorcontrib>Moncada-Velez, Marcela</creatorcontrib><creatorcontrib>Covill, Laura E</creatorcontrib><creatorcontrib>Zhang, Peng</creatorcontrib><creatorcontrib>Darazam, Ilad Alavi</creatorcontrib><creatorcontrib>Bastard, Paul</creatorcontrib><creatorcontrib>Bizien, Lucy</creatorcontrib><creatorcontrib>Bucciol, Giorgia</creatorcontrib><creatorcontrib>Enoksson, Sara Lind</creatorcontrib><creatorcontrib>Jouanguy, Emmanuelle</creatorcontrib><creatorcontrib>Karabela, Semsi Nur</creatorcontrib><creatorcontrib>Khan, Taushif</creatorcontrib><creatorcontrib>Kendir-Demirkol, Yasemin</creatorcontrib><creatorcontrib>Augusto Arias, Andres</creatorcontrib><creatorcontrib>Mansouri, Davood</creatorcontrib><creatorcontrib>Marits, Per</creatorcontrib><creatorcontrib>Marr, Nico</creatorcontrib><creatorcontrib>Migeotte, Isabelle</creatorcontrib><creatorcontrib>Moens, Leen</creatorcontrib><creatorcontrib>Ozcelik, Tayfun</creatorcontrib><creatorcontrib>Pellier, Isabelle</creatorcontrib><creatorcontrib>Sendel, Anton</creatorcontrib><creatorcontrib>Senoglu, Sevtap</creatorcontrib><creatorcontrib>Shahrooei, Mohammad</creatorcontrib><creatorcontrib>Smith, C.I. 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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.</description><issn>0022-1007</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>FZOIL</sourceid><recordid>eNqVjMsKwjAUBbNQsD7-IWuhcpO-cC0W3RbBZQjpLYnWtjRpa__eLPwAXZ2BGc6CBACchwwgW5G1tQ8AFsdJGpB7gbYzvXRtP9PRQ01NU6Fypm2sR9o6jf1kLFKNsnZ6pnp4Se8m47QPvDUOS3ot8oyWWBllsFHzliwrWVvcfXdD9vn5drqEz6HGYcRGlLaTCgUHkQAIxo8xFxkwSFm0IYefY-HeLvrr_QNAKlKP</recordid><startdate>20220615</startdate><enddate>20220615</enddate><creator>Campbell, Tessa Mollie</creator><creator>Liu, Zhiyong</creator><creator>Zhang, Qian</creator><creator>Moncada-Velez, Marcela</creator><creator>Covill, Laura E</creator><creator>Zhang, Peng</creator><creator>Darazam, Ilad Alavi</creator><creator>Bastard, Paul</creator><creator>Bizien, Lucy</creator><creator>Bucciol, Giorgia</creator><creator>Enoksson, Sara Lind</creator><creator>Jouanguy, Emmanuelle</creator><creator>Karabela, Semsi Nur</creator><creator>Khan, Taushif</creator><creator>Kendir-Demirkol, Yasemin</creator><creator>Augusto Arias, Andres</creator><creator>Mansouri, Davood</creator><creator>Marits, Per</creator><creator>Marr, Nico</creator><creator>Migeotte, Isabelle</creator><creator>Moens, Leen</creator><creator>Ozcelik, Tayfun</creator><creator>Pellier, Isabelle</creator><creator>Sendel, Anton</creator><creator>Senoglu, Sevtap</creator><creator>Shahrooei, Mohammad</creator><creator>Smith, C.I. 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Edvard</au><au>Vandernoot, Isabelle</au><au>Willekens, Karen</au><au>Yasar, Kadriye Kart</au><au>Bergman, Peter</au><au>Abel, Laurent</au><au>Cobat, Aurelie</au><au>Casanova, Jean-Laurent</au><au>Meyts, Isabelle</au><au>Bryceson, Yenan T</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Respiratory viral infections in otherwise healthy humans with inherited IRF7 deficiency</atitle><jtitle>JOURNAL OF EXPERIMENTAL MEDICINE</jtitle><date>2022-06-15</date><risdate>2022</risdate><volume>219</volume><issue>7</issue><issn>0022-1007</issn><abstract>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.</abstract><pub>ROCKEFELLER UNIV PRESS</pub><oa>free_for_read</oa></addata></record> |
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title | Respiratory viral infections in otherwise healthy humans with inherited IRF7 deficiency |
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