The first known human case of Trichinella pseudospiralis myositis is described. A 33 years old woman reported 5 years of relatively mild symptoms of tiredness, muscle fatigue and muscle pain after exercise. She had minimal proximal weakness. Creatinine kinase was significantly elevated, and muscle biopsy showed polymyositis and Trichinella larvae. Steroid treatment dramatically worsened the weakness. Treatment with albendazole led to complete resolution of symptoms and laboratory abnormalities. Diagnosis and identification of the parasite were based on the distinctive appearance of the unencapsulated larvae and their movement in fresh muscle, plus clinical and laboratory findings.