• Sanaz Lolachi, Sarah Morin, Matteo Coen, Kaveh Samii, Alexandra Calmy, and Jacques Serratrice.
• Division of General Internal Medicine, Department of Medicine.
• Medicine (Baltimore). 2020 Aug 7; 99 (32): e21570.

RationaleMacrophage activation syndrome (MAS) is a rare life-threatening condition characterized by cytokine-mediated tissue injury and multiorgan dysfunction.Patient ConcernsWe describe the unique case of young man who developed MAS as the sole manifestation of an otherwise paucisymptomatic severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection.DiagnosesClinical and biological criteria led to the diagnosis of MAS; cytokine profile was highly suggestive reverse transcription polymerase chain reaction for SARS-CoV-2 in nasopharyngeal swabs was negative, but serum anti-SARS-CoV-2 immunoglobulin A and immunoglobulin G resulted positive leading to the diagnosis of SARS-CoV-2 infection.InterventionsThe patient was treated with empiric antibiotic and hydroxychloroquine.OutcomesClinical improvement ensued. At follow-up, the patient is well.LessonSARS-CoV-2 infection may trigger develop life-threatening complications, like MAS. This can be independent from coronavirus disease 2019 gravity.

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