Genital infection by Schistosoma mansoni is usually misdiagnosed in individuals who reside in, or travel to endemic areas. We describe two cases of genital tumor associated with S. mansoni infection manifested by methrorragy. Surgical specimens revealed leiomyomas in both cases associated with S. mansoni. In one of them, granulomas were found in the ovary and in the other they were found in the uterine tube. Although none presented intestinal/hepatic disease, fecal egg excretion was detected in one. Both had elevated pretreatment antibody reactivity to S. mansoni antigen, but follow-up showed different outcomes. Schistosomiasis should be considered as a diagnosis in individuals with methrorragy residing in or having traveled to endemic areas. Since diagnosis follows genital amputation, and cure control is troublesome, improvement of diagnostic tools and follow-up markers are important priorities to decrease schistosomiasis morbidity.