Central nervous system (CNS) involvement may occur in chronic schistosomiasis. It can be produced by any Schistosome species but happens most frequently in chronic Schistosoma japonicum infection. CNS involvement by S. mansoni is relatively rare but it may occur by embolization of eggs or ectopic migration of adult worms. A case of cerebral schistosomiasis caused by S. mansoni in a 40-year-old man, who had worked in Yemen, is reported. Biopsies taken from the cerebellar vermis and the roof of the fourth ventricle, showed granulomatous inflammation due to eggs. S. mansoni was identified by stool examination and ELISA using serum and CSF. This is the first imported case of cerebral schistosomiasis in Korea.