SARS-CoV-2 infection in a pediatrics STAT1 GOF patient under Ruxolitinib therapy-a matter of balance?
| Autor: | Guillermo Martín Gutierrez, Beatriz de Felipe, Paloma Guisado Hernández, María Isabel Sánchez Codez, Peter Olbrich, José Manuel Lucena, María José Castro, Alicia Gutiérrez Valencia, Isabel Villaoslada, Mirella Gaboli, Olaf Neth, Pilar Blanco Lobo |
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| Přispěvatelé: | Instituto de Salud Carlos III, Junta de Andalucía, European Commission, Neth, Olaf [0000-0001-5018-0466], Neth, Olaf |
| Jazyk: | angličtina |
| Rok vydání: | 2021 |
| Předmět: |
0301 basic medicine
Ruxolitinib 2019-20 coronavirus outbreak Coronavirus disease 2019 (COVID-19) Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) Immunology Letter to Editor Severity of Illness Index 03 medical and health sciences 0302 clinical medicine Nitriles medicine Immunology and Allergy Humans Child Janus Kinases business.industry SARS-CoV-2 COVID-19 Interferon-Stimulated Gene Factor 3 COVID-19 Drug Treatment 030104 developmental biology Pyrimidines Mutation Pyrazoles Female business Humanities 030215 immunology medicine.drug |
| Zdroj: | Digital.CSIC. Repositorio Institucional del CSIC instname Journal of Clinical Immunology |
| DOI: | 10.1007/s10875-021-01081-9.pdf |
| Popis: | Recently, 94 inborn errors of immunity (IEI) patients suffering from COVID-19 have been described, overall demonstrating a mild phenotype [1] although more severe disease manifestations have been suggested for patients with alterations in the interferon (IFN) signaling pathway, including auto-antibodies against type I IFN [2]. Patients with STAT1 GOF mutations show a complex and often severe phenotype, combining an increased susceptibility of fungal, (myco-) bacterial and viral infections as well as autoimmune and autoinflammatory manifestations [3]. Characteristically, in response to type I and type II IFN stimulation, these patients show STAT1 hyperphosphorylation [3, 4]. Whether in the context of SARS-CoV-2 infection, the hyperactivation of the IFN-JAK1/2-STAT1 pathway would be protective (antiviral effect) or deleterious (hyperinflammation) is unclear. Ruxolitinib (a selective JAK1/2 inhibitor) has been successfully used in STAT1 GOF patients controlling many disease manifestations [5] and also resulted in improved pulmonary function and faster recovery from lymphopenia in previously healthy individuals suffering from severe COVID-19 [6]. This work was supported by the Instituto de Salud Carlos III, Madrid (Spain) [Sara Borrell, CD20/00124 to P.B.L, Juan Rodés JR18/00042 to P.O, FIS PI19/01471 to O.N] and the Consejería de Salud, Junta Andalucía [SA0051/2020 to O.N]. A.G-V was supported by the Instituto de Salud Carlos III, cofinanced by the European Development Regional Fund (“A way to achieve Europe”), Subprograma Miguel Servet (CP19/00159). |
| Databáze: | OpenAIRE |
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