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Introduction: Macrophage activation syndrome (MAS) is a rare complication of autoimmune diseases. The background of the diseases is immune system dysfunction. The disease is characterized by varying symptomatology and the different dynamics of accumulation of symptoms. Case report: A female patient diagnosed with adult-onset Still's disease was admitted to the hospital because of fever, weakness, skin rash and dyspepsia which had lasted for several days. Diagnostic tests revealed the presence of anti-nuclear antibodies, pancytopenia, hypertriglyceridemia, hyperferritinemia, hypofibrinogenemia, elevated activity of liver enzymes, and increased antibiotic therapy and intravenous pulses of methylprednisolone was unsuccessful and provided no improvement. On the basis of bone marrow cytological examination, the diagnosis of MAS was established. The patient was treated with dexamethasone and cyclosporine A with improvement. The patient was followed up in the rheumatology out-patient department for 20 months without the relapse of MAS. Conclusion: Macrophage activation syndrome is a rare, dangerous complication of autoimmune diseases, requiring rapid diagnosis and aggressive treatment. [ABSTRACT FROM AUTHOR]