The Authors report a case of paracoccidioidomycosis in a Surui Indian patient from the state of Rondonia, Brazilian Amazon. The subject is an adult male, having been diagnosed on the basis of mycologic, Serologic, and radiographic exams. The prescribed therapy was sulfametoxazol 800 mg associated with trimetoprim 160 mg every 12 hours. A revaluation of the patient conducted six months after the beginning of chemotherapy indicated overall improvement of his physical condition and of the radiologic picture, negativeness of the test of precipitin in capillary tube, and positiveness of the skin test with paracoccidioidin. The Authors also reviewed the cases of paracoccidioidomycosis described in the region, especially among the Surui Indian population.