• Sara Della Paolera, Erica Valencic, Elisa Piscianz, Valentina Moressa, Alberto Tommasini, Raffaella Sagredini, Valentina Kiren, Manola Comar, and Andrea Taddio.
• Department of Medicine, Surgery and Health Sciences, University of Trieste, Trieste, Italy.
• Front Pediatr. 2020 Jan 1; 8: 624248.

AbstractDuring COVID-19 outbreak, a large number of children with severe inflammatory disease has been reported. This condition, named Pediatric Multi-inflammatory Syndrome temporally associated with COVID-19 (PIMS-TS) or Multisystem Inflammatory Syndrome associated with Coronavirus Disease 2019 (MIS-C), shares some clinical features with Kawasaki disease and is frequently complicated by myocarditis or shock. It has been suggested that MIS-C belongs to the group of cytokine storm syndromes triggered by SARS-CoV-2 infection. So far, intravenous immunoglobulin (IVIG) and systemic glucocorticoids are the most common therapeutic approaches reported in this group of patients. However, the use of anakinra in patients with severe forms of COVID-19 is showing promising results. Here we reported two patients with multisystem inflammatory syndrome complicated with shock. Both the patients presented a poor response to IVIG and systemic glucocorticoids and received anakinra. Treatment with IL-1 receptor antagonist showed a rapid improvement of clinical conditions and biochemical analysis in both patients and demonstrated a good safety profile. Thus, we look forward for future controlled clinical trials with the aim to demonstrate the effectiveness of anakinra in patients with MIS-C and established precise criteria for its use.Copyright © 2021 Della Paolera, Valencic, Piscianz, Moressa, Tommasini, Sagredini, Kiren, Comar and Taddio.

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