Schistosomiasis is a neglected tropical disease that affects developing countries worldwide and is caused by several species of parasites from the Schistosoma genus. Chronic infection is characterized by the formation of granulomas around the parasite eggs, the leading cause of pathology. The hepatosplenic clinical form is one of the most common, but urogenital schistosomiasis is another relevant clinical presentation responsible for infertility in men and women. Inflammatory response, anatomical deformations, and endocrine/biochemical changes are involved in the development of infertility. Schistosome parasites can synthesize catechol estrogen-like molecules and affect the sexual hormone balance in their host. Here, we review many aspects of the pathology of urogenital schistosomiasis, specifically infertility, and point to the biochemical and endocrinal elements that must be investigated in the future.