Toxoplasmosis can cause polymyositis either by reactivation or by recent infection. Inconsistent response to antiprotozoal therapy has been the strongest argument against toxoplasmic polymyositis as a separate entity. We report a biopsy-proven case of toxoplasmic polymyositis in a cardiac transplant patient presenting with a severe proximal weakness, myopathic, electromyographic changes and ten-fold increase of anti-Toxoplasma antibodies. An early antiprotozoal therapy and plasmapheresis led to recovery. A review of previously reported cases of toxoplasmic polymyositis suggests that an early antiprotozoal therapy is the most important variable affecting the outcome of this disease. We propose that toxoplasmic polymyositis has two phases: acute, responsive to antiprotozoal therapy, and chronic, manifested by altered immune response requiring steroids. We suggest that all patients presenting with polymyositis should have serological tests for toxoplasmosis as a part of their initial evaluation and an early trial of antiprotozoal therapy in case of positive findings.