The second known human case of pulmonary anisakiasis is reported. A 22-years-old man living in Hyuga City, Miyazaki Prefecture, Japan, developed high fever, respiratory distress, and pleural effusion after consumption of raw fish. Although his total white blood cell count increased to approximately 10,000–20,000/mm3, eosinophilia was not observed. The total IgE level in his serum markedly increased up to 3,599 IU/ml. Since the patient was suspected to have a parasitic disease, immunoserologic tests were carried out. Screening tests using a multiple dot-enzyme-linked immunosorbent assay (ELISA) and an Ouchterlony double-diffusion test showed that his serum and pleural effusion had the strongest reactivity against crude antigen of Anisakis type I larvae, together with weak cross-reactivity against several other nematode antigens. Since extragastrointestinal anisakiasis was strongly suspected, this diagnosis was confirmed by a microplate-ELISA and Western blot analysis using a monoclonal antibody.