In this case report, we describe the rare situation of a patient with nonvariceal upper gastrointestinal bleeding induced by gastric and duodenal involvement of Schistosoma mansoni infection. In this unique case severe, recurrent upper gastrointestinal bleeding was induced by central ulcerations of gastric pseudopolypoid and duodenal polypoid lesions. However, very atypically, there were no signs of portal hypertension, coagulopathy, or variceal bleeding, and no macroscopic evidence of lower gastrointestinal tract involvement. Neither anti-ulcer therapy nor endoscopic hemostasis methods were effective in preventing recurrent bleeding episodes. Finally, typical histological and serological tests (positive for S. mansoni hemagglutination) led to the correct diagnosis, and the patient was completely cured by specific antischistosomal therapy.