Cystic echinococcosis or hydatid disease is an important public health issue, mainly in developing countries, due to its high prevalence. Echinococcus granulosus, a cyclophyllid cestode, the pathogenic parasite found in humans, their intermediate host in its way to the final host, the members of canidae family.

The main sites of infection in humans are the liver and the lungs. There have been recorded Rare locations such as the heart, spleen, muscles or retroperitoneal have also been recorded.

Case presentation: We present the case of a 29-year-old man, living in rural environment, who was admitted in our Clinic for a pelvic cystic tumor and intermittent ureterohydronephrosis. The blood work showed positive antibodies for Echinococcus granulosus. An urethrocystoscopy and the excision of the tumor were performed.

Conclusions: Although rare, the involvement of the male genitourinary tract in cystic echinococcosis is possible and can pose important diagnostic challenges.