The patient with polymyositis and antisynthetase syndrome treated with rituximab (Mabtera) is described. Rituximab was added to thehigh‑dose cyclophosphamide therapy due to an acute onset of the disease with a highly progressive interstitial lung disease and inability of the high‑dose corticosteroids therapy because of comorbidity. There was almost complete normalization of pulmonary and muscular pathological changes with more than 4‑fold decrease of anti‑Jo‑1 antibodies on the treatment. No

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... plications and no side effects during rituximab therapy were noted. This case report demonstrates the positive effect of rituximab in combination with high‑dose cyclophosphamide in the treatment of acute AS syndrome. The results published in literature are discussed.