Rare variants in Toll-like receptor 7 results in functional impairment and downregulation of cytokine-mediated signaling in COVID-19 patients.


Journal

Genes and immunity
ISSN: 1476-5470
Titre abrégé: Genes Immun
Pays: England
ID NLM: 100953417

Informations de publication

Date de publication:
02 2022
Historique:
received: 10 09 2021
accepted: 22 11 2021
revised: 11 11 2021
pubmed: 26 12 2021
medline: 1 3 2022
entrez: 25 12 2021
Statut: ppublish

Résumé

Toll-like receptors (TLR) are crucial components in the initiation of innate immune responses to a variety of pathogens, triggering the production of pro-inflammatory cytokines and type I and II interferons, which are responsible for innate antiviral responses. Among the different TLRs, TLR7 recognizes several single-stranded RNA viruses including SARS-CoV-2. We and others identified rare loss-of-function variants in X-chromosomal TLR7 in young men with severe COVID-19 and with no prior history of major chronic diseases, that were associated with impaired TLR7 signaling as well as type I and II IFN responses. Here, we performed RNA sequencing to investigate transcriptome variations following imiquimod stimulation of peripheral blood mononuclear cells isolated from patients carrying previously identified hypomorphic, hypofunctional, and loss-of-function TLR7 variants. Our investigation revealed a profound impairment of the TLR7 pathway in patients carrying loss-of-function variants. Of note, a failure in IFNγ upregulation following stimulation was also observed in cells harboring the hypofunctional and hypomorphic variants. We also identified new TLR7 variants in severely affected male patients for which a functional characterization of the TLR7 pathway was performed demonstrating a decrease in mRNA levels in the IFNα, IFNγ, RSAD2, ACOD1, IFIT2, and CXCL10 genes.

Identifiants

pubmed: 34952932
doi: 10.1038/s41435-021-00157-1
pii: 10.1038/s41435-021-00157-1
pmc: PMC8703210
doi:

Substances chimiques

Cytokines 0
TLR7 protein, human 0
Toll-Like Receptor 7 0
Toll-Like Receptor 8 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

51-56

Investigateurs

Francesca Montagnani (F)
Mario Tumbarello (M)
Ilaria Rancan (I)
Massimiliano Fabbiani (M)
Barbara Rossetti (B)
Laura Bergantini (L)
Miriana D'Alessandro (M)
Paolo Cameli (P)
David Bennett (D)
Federico Anedda (F)
Simona Marcantonio (S)
Sabino Scolletta (S)
Federico Franchi (F)
Maria Antonietta Mazzei (MA)
Susanna Guerrini (S)
Edoardo Conticini (E)
Luca Cantarini (L)
Bruno Frediani (B)
Danilo Tacconi (D)
Chiara Spertilli Raffaelli (CS)
Marco Feri (M)
Alice Donati (A)
Raffaele Scala (R)
Luca Guidelli (L)
Genni Spargi (G)
Marta Corridi (M)
Cesira Nencioni (C)
Leonardo Croci (L)
Gian Piero Caldarelli (GP)
Davide Romani (D)
Paolo Piacentini (P)
Maria Bandini (M)
Elena Desanctis (E)
Silvia Cappelli (S)
Anna Canaccini (A)
Agnese Verzuri (A)
Valentina Anemoli (V)
Manola Pisani (M)
Agostino Ognibene (A)
Alessandro Pancrazzi (A)
Maria Lorubbio (M)
Massimo Vaghi (M)
Antonella D'Arminio Monforte (A)
Federica Gaia Miraglia (FG)
Raffaele Bruno (R)
Marco Vecchia (M)
Massimo Girardis (M)
Sophie Venturelli (S)
Stefano Busani (S)
Andrea Cossarizza (A)
Andrea Antinori (A)
Alessandra Vergori (A)
Arianna Emiliozzi (A)
Stefano Rusconi (S)
Matteo Siano (M)
Arianna Gabrieli (A)
Agostino Riva (A)
Daniela Francisci (D)
Elisabetta Schiaroli (E)
Francesco Paciosi (F)
Andrea Tommasi (A)
Pier Giorgio Scotton (PG)
Francesca Andretta (F)
Sandro Panese (S)
Stefano Baratti (S)
Renzo Scaggiante (R)
Francesca Gatti (F)
Saverio Giuseppe Parisi (SG)
Francesco Castelli (F)
Eugenia Quiros-Roldan (E)
Melania Degli Antoni (MD)
Isabella Zanella (I)
Matteo Della Monica (M)
Carmelo Piscopo (C)
Mario Capasso (M)
Roberta Russo (R)
Immacolata Andolfo (I)
Achille Iolascon (A)
Giuseppe Fiorentino (G)
Massimo Carella (M)
Marco Castori (M)
Filippo Aucella (F)
Pamela Raggi (P)
Rita Perna (R)
Matteo Bassetti (M)
Antonio Di Biagio (A)
Maurizio Sanguinetti (M)
Luca Masucci (L)
Alessandra Guarnaccia (A)
Serafina Valente (S)
Oreste De Vivo (O)
Gabriella Doddato (G)
Mirjam Lista (M)
Giada Beligni (G)
Floriana Valentino (F)
Kristina Zguro (K)
Rossella Tita (R)
Annarita Giliberti (A)
Maria Antonietta Mencarelli (MA)
Caterina Lo Rizzo (CL)
Anna Maria Pinto (AM)
Francesca Ariani (F)
Laura Di Sarno (L)
Elena Bargagli (E)
Marco Mandalà (M)
Alessia Giorli (A)
Lorenzo Salerni (L)
Patrizia Zucchi (P)
Pierpaolo Parravicini (P)
Elisabetta Menatti (E)
Tullio Trotta (T)
Ferdinando Giannattasio (F)
Gabriella Coiro (G)
Fabio Lena (F)
Gianluca Lacerenza (G)
Cristina Mussini (C)
Enrico Martinelli (E)
Luisa Tavecchia (L)
Mary Ann Belli (MA)
Lia Crotti (L)
Gianfranco Parati (G)
Maurizio Sanarico (M)
Francesco Raimondi (F)
Filippo Biscarini (F)
Alessandra Stella (A)
Tiziana Bachetti (T)
Maria Teresa La Rovere (MT)
Serena Ludovisi (S)
Maurizio Bussotti (M)
Simona Dei (S)
Sabrina Ravaglia (S)
Rosangela Artuso (R)
Elena Andreucci (E)
Giulia Gori (G)
Angelica Pagliazzi (A)
Erika Fiorentini (E)
Antonio Perrella (A)
Francesco Bianchi (F)
Paola Bergomi (P)
Emanuele Catena (E)
Riccardo Colombo (R)
Sauro Luchi (S)
Giovanna Morelli (G)
Paola Petrocelli (P)
Sarah Iacopini (S)
Sara Modica (S)
Silvia Baroni (S)
Francesco Vladimiro Segala (FV)
Marco Falcone (M)
Giusy Tiseo (G)
Chiara Barbieri (C)
Tommaso Matucci (T)
Davide Grassi (D)
Claudio Ferri (C)
Franco Marinangeli (F)
Francesco Brancati (F)
Antonella Vincenti (A)
Valentina Borgo (V)
Stefania Lombardi (S)
Mirco Lenzi (M)
Massimo Antonio Di Pietro (MA)
Francesca Vichi (F)
Benedetta Romanin (B)
Letizia Attala (L)
Cecilia Costa (C)
Andrea Gabbuti (A)
Roberto Menè (R)
Marta Colaneri (M)
Patrizia Casprini (P)
Giuseppe Merla (G)
Gabriella Maria Squeo (GM)
Marcello Maffezzoni (M)

Informations de copyright

© 2021. The Author(s).

Références

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Auteurs

Stefania Mantovani (S)

Division of Clinical Immunology and Infectious Diseases, Department of Medicine, Fondazione IRCCS Policlinico San Matteo, Pavia, Italy.

Sergio Daga (S)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Chiara Fallerini (C)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Margherita Baldassarri (M)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Elisa Benetti (E)

Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Nicola Picchiotti (N)

Department of Mathematics, University of Pavia, Pavia, Italy.
University of Siena, DIISM-SAILAB, Siena, Italy.

Francesca Fava (F)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.
Genetica Medica, Azienda Ospedaliero-Universitaria Senese, Siena, Italy.

Anna Gallì (A)

Department of Hematology Oncology, IRCCS Fondazione Policlinico San Matteo, Pavia, Italy.

Silvia Zibellini (S)

Department of Hematology Oncology, IRCCS Fondazione Policlinico San Matteo, Pavia, Italy.

Mirella Bruttini (M)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.
Genetica Medica, Azienda Ospedaliero-Universitaria Senese, Siena, Italy.

Maria Palmieri (M)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Susanna Croci (S)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Sara Amitrano (S)

Genetica Medica, Azienda Ospedaliero-Universitaria Senese, Siena, Italy.

Diana Alaverdian (D)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Katia Capitani (K)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.
Core Research Laboratory, ISPRO, Florence, Italy.

Simone Furini (S)

Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Francesca Mari (F)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.
Genetica Medica, Azienda Ospedaliero-Universitaria Senese, Siena, Italy.

Ilaria Meloni (I)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Elisa Frullanti (E)

Medical Genetics, University of Siena, Siena, Italy.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy.

Mario U Mondelli (MU)

Division of Clinical Immunology and Infectious Diseases, Department of Medicine, Fondazione IRCCS Policlinico San Matteo, Pavia, Italy. m.mondelli@smatteo.pv.it.
Department of Internal Medicine and Therapeutics, University of Pavia, Pavia, Italy. m.mondelli@smatteo.pv.it.

Alessandra Renieri (A)

Medical Genetics, University of Siena, Siena, Italy. alessandra.renieri@unisi.it.
Med Biotech Hub and Competence Center, Department of Medical Biotechnologies, University of Siena, Siena, Italy. alessandra.renieri@unisi.it.
Genetica Medica, Azienda Ospedaliero-Universitaria Senese, Siena, Italy. alessandra.renieri@unisi.it.

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