The neurological involvement in Manson's Schistosomiasis is rarely reported in the literature. The authors describe a case of a 23 years old patient born in São Francisco-MG. She presented with headache, vomiting, fever and stupor. The neurological examination showed stiffness of the neck, paresis with bilateral muscular hypertony in the upper members, and loss of motricity in inferior members. There was no sign of hepatosplenomegaly. Cerebrospinal fluid examination showed: a decrease in the glucose content (28 mg/dl), increase in the protein content (113 mg/dl) and an increase in cell number (949 cells/mm3) with predominance of neutrophils (70%) and presence of eosinophils (9%); the indirect immunofluorescence test using samples of mouse's liver with periovular granuloma was reactive (IgG 1/16; IgM 1/4); the search for acid-fast bacilli and fungi, as well as the culture for acid fast bacilli (after 60 days) and the cysticercosis immunological tests were negative. The computerized tomography of the brain and spinal cord did not show abnormalites. Fecal smear examination showed mature eggs of S. mansoni. Treatment was made with corticosteroids and praziquantel (60 mg/kg/day; 7 days), and the cerebrospinal fluid alterations remitted. However, severe neurological sequels (paraplegia and neurogenic bladder) remained.