A 61-year-old man was admitted to our hospital with a 6-month history of productive cough. He, along with his wife, had been involved with Shiitake mushroomcultures for a period of 12 years. On admission, chest radiography showed bilateral fine-nodular shadowand CTscans showed reticulonodular opacities and a ground-glass appearance predominantly in the subpleural area in both lungs, and a mass in the left S6. Resected pathological specimens obtained by left lower lobectomy revealed lung adenosquamous carcinoma (stage IB), interstitial changes accompanied with lymphocyte proliferation and fibrosis, and granuloma with giant cells. Serumprecipitins for Shiitake mushroomantigens were positive. The productive cough improved after the hospital admission and occurred again whenhe returned to work with the Shiitake mushroomproduction. Therefore, chronic hypersensitivity pneumonitis (HP) caused by Shiitake mushroomspores was diagnosed. Moreover, his wife was found to have HP caused by mushrooms at this time. There are only two previous reports of chronic HP caused by Shiitake mushroom in Japan, and this is the first case of chronic HP associated with lung cancer.