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Publication Details
AFRICAN RESEARCH NEXUS
SHINING A SPOTLIGHT ON AFRICAN RESEARCH
general
Inborn errors of OAS-RNase L in SARS-CoV-2-related multisystem inflammatory syndrome in children
Science, Volume 379, No. 6632, Article eabo3627, Year 2023
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Description
Multisystem inflammatory syndrome in children (MIS-C) is a rare and severe condition that follows benign COVID-19. We report autosomal recessive deficiencies of OAS1, OAS2, or RNASEL in five unrelated children with MIS-C. The cytosolic double-stranded RNA (dsRNA)-sensing OAS1 and OAS2 generate 2'-5'-linked oligoadenylates (2-5A) that activate the single-stranded RNA-degrading ribonuclease L (RNase L). Monocytic cell lines and primary myeloid cells with OAS1, OAS2, or RNase L deficiencies produce excessive amounts of inflammatory cytokines upon dsRNA or severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) stimulation. Exogenous 2-5A suppresses cytokine production in OAS1-deficient but not RNase L-deficient cells. Cytokine production in RNase L-deficient cells is impaired by MDA5 or RIG-I deficiency and abolished by mitochondrial antiviral-signaling protein (MAVS) deficiency. Recessive OAS-RNase L deficiencies in these patients unleash the production of SARS-CoV-2-triggered, MAVS-mediated inflammatory cytokines by mononuclear phagocytes, thereby underlying MIS-C. © 2023 American Association for the Advancement of Science. All rights reserved.
Authors & Co-Authors
Ogishi, Masato
United States, New York
Rockefeller University
Talouarn, Estelle
France, Paris
L'institut Des Maladies Génétiques Imagine
Rinchai, Darawan
United States, New York
Rockefeller University
Zhang, Peng
United States, New York
Rockefeller University
Jordán, Iolanda
Spain, Esplugues de Llobregat
Hospital Sant Joan de Déu Barcelona
Spain, Madrid
Instituto de Salud Carlos Iii
Spain, Barcelona
Universitat de Barcelona
Spain, Barcelona
Institut de Recerca Sant Joan de Déu
Bozdemir, Şefika Elmas
Turkey, Bursa
Bursa City Hospital
Bizien, Lucy
France, Paris
L'institut Des Maladies Génétiques Imagine
Lei, Weite
United States, New York
Rockefeller University
Hasan, Milena
France, Paris
Institut Pasteur, Paris
Libri, Valentina
France, Paris
Institut Pasteur, Paris
Hoffmann, Hans Heinrich
United States, New York
Rockefeller University
Michailidis, Eleftherios
United States, New York
Rockefeller University
United States, Atlanta
Emory University
Moreews, Marion
France, Lyon
Centre International de Recherche en Infectiologie
Seeleuthner, Yoann
France, Paris
L'institut Des Maladies Génétiques Imagine
Bilgüvar, Kaya
United States, New Haven
Yale School of Medicine
Turkey, Istanbul
Acibadem Mehmet Ali Aydinlar Universitesi
Mane, Shrikant M.
United States, New Haven
Yale School of Medicine
Flores, Carlos M.
Spain, Santa Cruz de Tenerife
Hospital Universitario Nuestra Senora de Candelaria
Spain, Madrid
Centro de Investigación Biomédica en Red de Enfermedades Respiratorias
Zhang, Yu
United States, Bethesda
National Institutes of Health Nih
Arias, Andrés Augusto
United States, New York
Rockefeller University
Colombia, Medellin
Universidad de Antioquia
Schlüter, Agatha
Spain, Madrid
Centro de Investigación Biomédica en Red de Enfermedades Raras
Milisavljevic, Baptiste
United States, New York
Rockefeller University
Bigio, Benedetta
United States, New York
Rockefeller University
Le Voyer, Tom
France, Paris
L'institut Des Maladies Génétiques Imagine
Gervais, Adrian
France, Paris
L'institut Des Maladies Génétiques Imagine
Moncada-Vélez, Marcela
United States, New York
Rockefeller University
Pala, Francesca
United States, Bethesda
National Institutes of Health Nih
Lazarov, Tomi
United States, New York
Memorial Sloan-kettering Cancer Center
Levy, Romain
France, Paris
L'institut Des Maladies Génétiques Imagine
Neehus, Anna Lena
France, Paris
L'institut Des Maladies Génétiques Imagine
Rosain, Jérémie
France, Paris
L'institut Des Maladies Génétiques Imagine
Peel, Jessica N.
United States, New York
Rockefeller University
Belkaya, Serkan
Turkey, Ankara
Bilkent Üniversitesi
Lorenzo, Lazaro
United States, New York
Rockefeller University
Anton, Jordi
Spain, Barcelona
Universitat de Barcelona
Spain, Esplugues de Llobregat
Hospital Sant Joan de Déu Barcelona
Spain, Barcelona
Institut de Recerca Sant Joan de Déu
Delafontaine, Selket
Belgium, Leuven
Ku Leuven– University Hospital Leuven
Belgium, Leuven
Ku Leuven
Toubiana, Julie
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
France, Paris
Institut Pasteur, Paris
Fumadó, Victòria Pérez
Spain, Barcelona
Universitat de Barcelona
Spain, Esplugues de Llobregat
Hospital Sant Joan de Déu Barcelona
Spain, Barcelona
Institut de Recerca Sant Joan de Déu
Rozenberg, Flore
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Geissmann, Frédéric
United States, New York
Memorial Sloan-kettering Cancer Center
Lebon, Pierre
Unknown Affiliation
Weiss, Susan Reich
United States, Philadelphia
University of Pennsylvania
Bonnet, Damien
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Duval, Xavier
France, Paris
Hôpital Bichat-claude-bernard Ap-hp
France, Paris
Université Paris Cité
France, Paris
Infection, Anti-microbien, Modélisation, Évolution
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Pan-Hammarström, Qiang
Sweden, Stockholm
Karolinska Institutet
Planas, Anna M.
Spain, Madrid
Consejo Superior de Investigaciones Científicas
Spain, Barcelona
Institut D'investigacions Biomèdiques August pi I Sunyer - Idibaps
Meyts, Isabelle
Belgium, Leuven
Ku Leuven
Haerynck, Filomeen
Belgium, Ghent
Universitair Ziekenhuis Gent
Pujol, Aurora
Spain, Barcelona
Institució Catalana de Recerca I Estudis Avançats
Spain, Madrid
Centro de Investigación Biomédica en Red de Enfermedades Raras
Sancho-Shimizu, Vanessa
United Kingdom, London
Imperial College London
Dalgard, Clifton L.
United States, Bethesda
Uniformed Services University of the Health Sciences
Bustamante, Jacinta C.
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Puel, Anne
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Boisson-Dupuis, Stéphanie
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Boisson, Bertrand
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Maniatis, Tom
United States, New York
New York Genome Center
Zhang, Qian
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Bastard, Paul
France, Paris
L'institut Des Maladies Génétiques Imagine
Spain, Barcelona
Universitat de Barcelona
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Notarangelo, Luigi Daniele
United States, Bethesda
National Institutes of Health Nih
Béziat, Vivien
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Pérez de Diego, Rebeca
Spain, Madrid
Hospital Universitario la Paz
Rodríguez-Gallego, Carlos
Spain, Santa Maria de Guia de Gran Canaria
Universidad Fernando Pessoa Canarias
Spain, Las Palmas de Gran Canaria
Hospital de Gran Canaria Dr. Negrin
Su, Helen C.
United States, Bethesda
National Institutes of Health Nih
Lifton, Richard P.
United States, New Haven
Yale School of Medicine
United States, New York
Rockefeller University
Jouanguy, Emmanuelle
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Cobat, Aurélie
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Alsina, Laia
Spain, Barcelona
Universitat de Barcelona
Spain, Barcelona
Institut de Recerca Sant Joan de Déu
Spain, Esplugues de Llobregat
Hospital Sant Joan de Déu Barcelona
Keleş, Sevgi
Turkey, Meram
Necmettin Erbakan Üniversitesi
Haddad, Elie
Canada, Montreal
University of Montreal
Abel, Laurent
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Belot, Alexandre
France, Lyon
Centre International de Recherche en Infectiologie
France, Lyon
Chu de Lyon
Quintana-Murci, Lluís
France, Paris
Cnrs Centre National de la Recherche Scientifique
France, Paris
Collège de France
Rice, Charles M.
United States, New York
Rockefeller University
Zhang, Shenying
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
Casanova, Jean Laurent
United States, New York
Rockefeller University
France, Paris
L'institut Des Maladies Génétiques Imagine
France, Paris
Hôpital Necker Enfants Malades
United States, Chevy Chase
Howard Hughes Medical Institute
France, Paris
Inserm
Behillil, Sylvie
France, Paris
Institut Pasteur, Paris
Burdet, Charles
France, Paris
Infection, Anti-microbien, Modélisation, Évolution
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Charpentier, Charlotte
France, Paris
Infection, Anti-microbien, Modélisation, Évolution
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Descamps, Diáne
France, Paris
Infection, Anti-microbien, Modélisation, Évolution
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Ecobichon, Jean Luc
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Enouf, Vincent V.E.
France, Paris
Institut Pasteur, Paris
Houhou-Fidouh, Nadhira
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Lina, Bruno
France, Lyon
Centre International de Recherche en Infectiologie
Lucet, Jean Christophe
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Manchon, Pauline
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Tubiana, Sarah
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Van Der Werf, Sylvie
France, Paris
Institut Pasteur, Paris
Visseaux, Benoit
France, Paris
Infection, Anti-microbien, Modélisation, Évolution
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Yazdanpanah, Yazdan
France, Paris
Infection, Anti-microbien, Modélisation, Évolution
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Waucquier, Nawal
Unknown Affiliation
Deplanque, D.
Unknown Affiliation
Djossou, Félix
Unknown Affiliation
Demar, Magalie Pierre
Unknown Affiliation
Bruneau, Leá
France, Paris
Inserm
Gérardin, Patrick
France, Paris
Inserm
Lainé, Fabrice
France, Rennes
Centre D'investigation Clinique de Rennes
Malvy, Denis Jean Marie
France, Talence
Centre Hospitalier Universitaire de Bordeaux
Nguyen, Duc Bang
France, Talence
Centre Hospitalier Universitaire de Bordeaux
Pistone, Thierry
France, Talence
Centre Hospitalier Universitaire de Bordeaux
Chirouze, Catherine
France, Besancon
Centre Hospitalier Universitaire de Besançon
Bouiller, Kevin
France, Besancon
Centre Hospitalier Universitaire de Besançon
Desmarets, Maxime
France, Paris
Inserm
Lefèvre, Benjamin
France, Nancy
Chu de Nancy
Jeulin, Hélène
France, Vandoeouvre-les-nancy
Hôpital Brabois Adultes
Legrand, Karine
France, Paris
Inserm
Lomazzi, Sandra
France, Nancy
Chu de Nancy
Tardy, Bernhard
France, Paris
Inserm
Gagneux-Brunon, Amandine
France, Saint-etienne
Centre Hospitalier Universitaire de Saint Etienne
Bertholon, Frédérique
France, Saint-etienne
Centre Hospitalier Universitaire de Saint Etienne
Botelho-Nevers, Elisabeth
France, Saint-etienne
Centre Hospitalier Universitaire de Saint Etienne
Couffin-Cadièrgues, Sandrine
France, Paris
Inserm
Esperou, Helene
France, Paris
Inserm
Abolhassani, Hassan
Sweden, Stockholm
Karolinska Institutet
Aguilera-Albesa, Sergio
Unknown Affiliation
Aïuti, Alessandro
Unknown Affiliation
AlKhater, Suzan A.
Saudi Arabia, Dammam
Imam Abdulrahman Bin Faisal University
Allende, Luís Miguel
Spain, Madrid
Hospital Universitario 12 de Octubre
Amenzoui, Naïma
Morocco, Casablanca
Hassan Ii University of Casablanca
Arkin, Lisa M.
United States, Madison
University of Wisconsin-madison
Aubart, Mélodie
France, Paris
L'institut Des Maladies Génétiques Imagine
France, Paris
Ap-hp Assistance Publique - Hopitaux de Paris
Aytekin, Caner
Turkey, Ankara
Dr. Sami Ulus Maternity and Children's Health and Diseases Training and Research Hospital
Aytekin, Gökhan
Turkey, Konya
Konya City Hospital
Biggs, Catherine M.
Canada, Vancouver
The University of British Columbia
Canada, Vancouver
Bc Children's Hospital Research Institute
Blázquez-Gamero, Daniel
Spain, Madrid
Hospital Universitario 12 de Octubre
Bloomfield, Markéta
Czech Republic, Prague
Charles University
Bogunovic, Dusan
United States, New York
Icahn School of Medicine at Mount Sinai
Bondarenko, Anastasiia V.
Ukraine, Kyiv
Shupyk National Healthcare University of Ukraine
Borghesi, Alessandro
Italy, Pavia
Fondazione Irccs Policlinico San Matteo
Switzerland, Lausanne
École Polytechnique Fédérale de Lausanne
Bousfiha, Ahmed Aziz
Morocco, Casablanca
Centre Hospitalier Universitaire Ibn Rochd
Boyarchuk, Oksana R.
Ukraine, Ternopil
I. Horbachevsky Ternopil National Medical University
Brodin, Petter
Sweden, Solna
Science for Life Laboratory
Bryceson, Yenan Troi
Sweden, Stockholm
Karolinska Institutet
Bucciol, Giorgia
Belgium, Leuven
Ku Leuven– University Hospital Leuven
Calcaterra, Valeria
Italy, Milan
Ospedale Dei Bambini Vittore Buzzi
Italy, Pavia
Università Degli Studi Di Pavia
Casari, Giorgio
Unknown Affiliation
Cavalcanti, André De Souza
Brazil, Recife
Universidade Federal de Pernambuco
Chrousos, George P.
Greece, Athens
National and Kapodistrian University of Athens
Colobran Oriol, Roger
Spain, Barcelona
Vall D'hebron Institut de Recerca
Condino‑Neto, Antonio A.
Brazil, Sao Paulo
Universidade de São Paulo
Conti, Francesca
Italy, Bologna
Alma Mater Studiorum Università Di Bologna
Cooper, Megan A.
United States, St. Louis
Washington University in St. Louis
Cyrus, Cyril
Saudi Arabia, Dammam
Imam Abdulrahman Bin Faisal University
El Zein, Loubna
Lebanon, Beirut
Université Libanaise
Elnagdy, Marwa H.
Egypt, Mansoura
Mansoura University
Emiroǧlu, Melike Keser
Turkey, Selçuklu
Selçuk Üniversitesi
Erdeniz, Emine Hafize
Turkey, Samsun
Ondokuz Mayis Üniversitesi
Fabi, Marianna
Italy, Bologna
Irccs Azienda Ospedaliero-universitaria Di Bologna
Baris-Feldman, Hagit
Israel, Tel Aviv-yafo
Tel Aviv Sourasky Medical Center
Fellay, Jacques
Switzerland, Lausanne
École Polytechnique Fédérale de Lausanne
Switzerland, Lausanne
Université de Lausanne Unil
Switzerland, Lausanne
Sib Swiss Institute of Bioinformatics
Gülhan, Belgin
Turkey, Ankara
Ankara City Hospital
Gut, Marta
Spain, Barcelona
Barcelona Institute of Science and Technology Bist
Halwani, Rabih
United Arab Emirates, Sharjah
University of Sharjah
Hammarström, Lennart L.G.
Sweden, Stockholm
Karolinska Universitetssjukhuset
Hatipoǧlu, Nevin
Turkey, Istanbul
Bak?rkoy Dr. Sadi Konuk Egitim ve Arast?rma Hastanesi
Heath, James R.
United States, Seattle
Institute for Systems Biology
Henrickson, Sarah E.
United States, Philadelphia
University of Pennsylvania
United States, Philadelphia
The Children's Hospital of Philadelphia
Hoste, Levi
Belgium, Ghent
Universitair Ziekenhuis Gent
Hsieh, Elena Wen Yuan
United States, Aurora
The Children's Hospital, Aurora
Itan, Yuval
United States, New York
Icahn School of Medicine at Mount Sinai
KandEmİr, Bahar
Turkey, Meram
Necmettin Erbakan Üniversitesi
Karbuz, Adem
Turkey, Istanbul
Prof. Dr. Cemil Tascioglu City Hospital
Kasapcopur, Ozgur
Turkey, Istanbul
Istanbul University-cerrahpasa
Hansen, Stella Kim
United States, New York
Suny Downstate Health Sciences University
Klocperk, Adam
Czech Republic, Prague
Charles University
Lau, Yu Lung
Hong Kong, Hong Kong
The University of Hong Kong
Lebl, Jan
Czech Republic, Prague
Charles University
Lucas, Carrie L.
United States, New Haven
Yale University
Mentis, Alexios Fotios A.
Greece, Athens
National and Kapodistrian University of Athens
Pato, Michèle T.
United States, New York
Suny Downstate Health Sciences University
Milner, Joshua D.
United States, New York
Columbia University
Mogensen, Trine Hyrup
Denmark, Aarhus
Aarhus Universitet
Nepesov, Serdar
Turkey, Istanbul
İstanbul Medipol Üniversitesi
Neves, João Farela
Portugal, Lisbon
Hospital de Dona Estefânia
Ng, Lisa F.P.
Singapore, Singapore City
Agency for Science, Technology and Research, Singapore
Novelli, Antonio
Italy, Rome
Irccs Ospedale Pediatrico Bambino Gesù
Novelli, Giuseppe
Italy, Rome
Università Degli Studi Di Roma Tor Vergata
Öz, Fatma Nur
Turkey, Ankara
Dr. Sami Ulus Maternity and Children's Health and Diseases Training and Research Hospital
Okada, Satoshi
Japan, Hiroshima
Hiroshima University Graduate School of Biomedical and Health Sciences
Ouair, Hind
Morocco, Casablanca
Centre Hospitalier Universitaire Ibn Rochd
Özkaya-Parlakay, Aslinur Nur
Turkey, Ankara
Ankara Yildirim Beyazit University
Pato, Carlos N.
Turkey, Eskisehir
Eskişehir Osmangazi Üniversitesi
Paz-Artal, Estela
Spain, Madrid
Hospital Universitario 12 de Octubre
Pelham, Simon J.
United States, New York
Rockefeller University
Pellier, Isabelle
France, Angers
Chu Angers
Philippot, Quentin
France, Paris
L'institut Des Maladies Génétiques Imagine
Planas-Serra, Laura
Spain, Madrid
Centro de Investigación Biomédica en Red de Enfermedades Raras
Prando, Carolina Cardoso De Mello
Brazil, Curitiba
Faculdades Pequeno Príncipe
RENIA, LAURENT
Singapore, Singapore City
Agency for Science, Technology and Research, Singapore
Singapore, Singapore City
Nanyang Technological University
Rivière, Jacques G.
Spain, Barcelona
Vall D'hebron Institut de Recerca
Salifu, Moro O.
United States, New York
Suny Downstate Health Sciences University
Schmitt, Erica G.
United States, St. Louis
Washington University in St. Louis
Šedivá, Anná
Czech Republic, Prague
Charles University
Slaby, Ondřej
Czech Republic, Brno
Masaryk University
Sobh, Ali
Egypt, Mansoura
Mansoura University Children's Hospital
Solé-Violán, Jordi
Spain, Las Palmas de Gran Canaria
Hospital de Gran Canaria Dr. Negrin
Spain, Santa Maria de Guia de Gran Canaria
Universidad Fernando Pessoa Canarias
Spain, Madrid
Instituto de Salud Carlos Iii
Soler-Palacín, P.
Spain, Barcelona
Vall D'hebron Institut de Recerca
de Somer, Lien
Belgium, Leuven
Ku Leuven– University Hospital Leuven
Sözeri, Betül Y.
Turkey, Istanbul
University of Health Sciences
Spaan, András N.
United States, New York
Rockefeller University
Netherlands, Utrecht
University Medical Center Utrecht
Stepanovskyy, Yuriy
Ukraine, Kyiv
Shupyk National Healthcare University of Ukraine
Tangye, Stuart G.
Australia, Sydney
Garvan Institute of Medical Research
Australia, Sydney
Unsw Sydney
Tanír, Gön̈ul
Turkey, Ankara
Dr. Sami Ulus Maternity and Children's Health and Diseases Training and Research Hospital
Thorball, Christian W.
Switzerland, Lausanne
Université de Lausanne Unil
Hançerli Törün, Selda
Turkey, Istanbul
Istanbul Üniversitesi
Turvey, Stuart E.
Canada, Vancouver
The University of British Columbia
Canada, Vancouver
Bc Children's Hospital Research Institute
Abou Tayoun, Ahmad N.
United Arab Emirates, Dubai
Al Jalila Children's Specialty Hospital
United Arab Emirates, Dubai
Mohammed Bin Rashid University of Medicine and Health Sciences
Ramaswamy, Sathishkumar
United Arab Emirates, Dubai
Al Jalila Children's Specialty Hospital
Uddin, Mohammed Jashim
United Arab Emirates, Dubai
Mohammed Bin Rashid University of Medicine and Health Sciences
Canada, Toronto
Genomearc Inc.
Vermeulen, François L.
Belgium, Leuven
Ku Leuven– University Hospital Leuven
Vinh, Donald C.
Canada, Montreal
Centre Universitaire de Santé Mcgill
Volokha, Alla P.
Ukraine, Kyiv
Shupyk National Healthcare University of Ukraine
von Bernuth, Horst
Germany, Berlin
Charité – Universitätsmedizin Berlin
Germany, Berlin
Labor Berlin Gmbh
Germany, Berlin
Berliner Institut Für Gesundheitsforschung
Wouters, Carine Helena P.
Belgium, Leuven
Ku Leuven– University Hospital Leuven
Zatz, Mayana
Brazil, Sao Paulo
Universidade de São Paulo
Zawadzki, Pawel
Unknown Affiliation
Zuccotti, Gianvincenzo Vincenzo
Italy, Milan
Ospedale Dei Bambini Vittore Buzzi
Italy, Milan
Università Degli Studi Di Milano
Statistics
Citations: 33
Authors: 215
Affiliations: 155
Identifiers
Doi:
10.1126/science.abo3627
ISSN:
00368075
Research Areas
Cancer
Covid
Maternal And Child Health