Case report of a patient recently arrived from Pernambuco (a region where schistosomiasis is endemic) with a very suggestive history of bilharzial contamination, presenting hepatosplenomegalia and a neurological picture including generalized weakness, specially in the lower limbs, absence of deep reflexes, hypotonus, tactile and thermalgesic hypesthesia in the hands and lower limbs, and marked impairment of proprioception in the distal segments of all limbs. Ova of Schistosoma monsoni were found in the feces. Positive intradermic reaction and complement fixation test in the blood. The assumption of schistosomiasis of the spinal cord was strengthened by a meningo-parenchymatous type of change in the cerebrospinal fluid, including definite pleocytosis with eosinophilia, and also by a strongly positive complement fixation test in the fluid. The patient died before anti-monial treatment could be started. The post mortem study revealed changes which usually occur in visceral schistosomiasis, but as ova were not found in the tissues, the diagnosis was maintained if not confirmed. The study of the nervous system showed normality of the brain; in the spinal cord it was found chronic leptomeningitis, radiculitis, bilateral degeneration of roots in the cauda equina, and secondary bilateral partial degeneration of the fasciculus gracilis.