Schistosomiasis is a parasitic disease reported in 78 countries, with an additional recent outbreak in Corsica. Generally, Schistosoma haematobium causes urogenital problems, whereas S. mansoni, S. japonicum, S. mekongi, S. guineensis, and S. intercalatum generate intestinal symptoms. Occasionally, ectopic tissue tropisms and infections by parasites resulting from hybridization occur. Geographical distribution and transmission of Schistosoma species depend on the presence of suitable intermediate snail hosts to complete the life cycle. Here we report on an unusual case of urogenital schistosomiasis in a Dominican adult male, living in Spain, with no history of visiting a known endemic area.