Urinary schistosomiasis is an endemic disease mainly distributed in most of Africa and Southwest Asia, and caused by infestation with Schistosoma haematobium. The case presented here was a 33-year-old Japanese male who had suffered from schistosomal infestation in the endemic area, and diagnosed as urinary schistosomiasis by detection of Schistosoma haematobium ova in urine and specimens obtained by mucosal biopsy of the bladder. Radiographic and endoscopic examinations revealed neither obstructive uropathy nor urothelial malignancy. The patient underwent treatment with antimonyl sodium tartrate (Stibnal), and the disease is under good control.