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DOI 10.1007/s10875-024-01840-4

Медицинская библиотека · Journal of clinical immunology · 2024

Inborn Error of WAS Presenting with SARS-CoV-2-Related Multisystem Inflammatory Syndrome in Children.

Drago Enrico, Fioredda Francesca, Penco Federica, Prigione Ignazia, Bertoni Arinna, Del Zotto Genny, Bocca Paola, Massaccesi Erika, Lanciotti Marina, Moratto Daniele, Thurner Lorenz, Caorsi Roberta, Gattorno Marco, Volpi Stefano

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Аннотация

Multisystem inflammatory syndrome in children (MIS-C) has been reported in patients with inborn errors of immunity (IEI), providing insights into disease pathogenesis. Here, we present the first case of MIS-C in a child affected by Wiskott-Aldrich syndrome (WAS) gene mutation, elucidating underlying predisposing factors and the involved inflammatory pathways. Genetic analysis revealed a frameshift truncating variant in the WAS gene, resulting in WAS protein expression between mild and severe forms, despite a clinical phenotype resembling X-linked thrombocytopenia (XLT). IL-1β secretion by LPS-stimulated peripheral blood mononuclear cells from patient during MIS-C was lower compared to healthy subjects but increased during follow-up. Conversely, the percentage of ASC (apoptosis-associated speck-like protein containing a CARD) specks in the patient's circulating monocytes during the acute phase was higher than in healthy subjects. The type I interferon (IFN) signature during MIS-C was normal, in contrast to the raised IFN signature measured far from the acute event. This case supports the association of IEI with MIS-C, potentially linked to delayed immune responses to SARS-CoV-2. The XLT phenotype underlies a subclinical immunodysregulation involving the NLRP3 inflammasome and the type-I IFN response.

Для цитирования: Drago Enrico, Fioredda Francesca, Penco Federica, Prigione Ignazia, Bertoni Arinna, Del Zotto Genny, Bocca Paola, Massaccesi Erika, Lanciotti Marina, Moratto Daniele, Thurner Lorenz, Caorsi Roberta, Gattorno Marco, Volpi Stefano. Inborn Error of WAS Presenting with SARS-CoV-2-Related Multisystem Inflammatory Syndrome in Children.. Journal of clinical immunology. 2024. DOI: 10.1007/s10875-024-01840-4.

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