During the current SARS-CoV-2 pandemic, a novel syndrome termed "
multisystem inflammatory syndrome in children" (MIS-C) has emerged. MIS-C was linked to
COVID-19 and shared some features with
Kawasaki disease and
Toxic Shock Syndrome, with a common pathogenetic substrate of hyperinflammation and
cytokine storm. Lately, MIS was also described in adults (≥21 years of age) and named "
MIS-A". There is no consensus about the treatment of
MIS-A; successful use of
glucocorticoids and
immunoglobulins has been reported in case series, but more solid evidence is lacking. Furthermore, the role of
biologic agents with proven benefits against
COVID-19, MIS-C, or
Kawasaki disease is still unexplored. In this report, we detail the clinical picture and the diagnostic process that led to the diagnosis of
MIS-A in a 27-year-old man, focusing on its treatment with
anakinra and
glucocorticoids, which resulted in full recovery. To our knowledge, this is the first report of the successful use of
anakinra for
MIS-A, a
drug that has already proven useful in the treatment of refractive cases of MIS-C.
Anakinra may also play a pivotal role for the treatment of
MIS-A.