We report on a case of prostate multilocular cystadenoma presenting with primary infertility and obstructive azoospermia. Our patient is a 36-year-old presenting with primary infertility in addition to mild deep pelvic pain. Semen analysis revealed azoospermia with positive fructose. His prostate-specific antigen was 0.7 ng/dL and his imaging revealed a large multilocular cystic mass with multiple internal enhancing septa. Transrectal ultrasonography-guided aspiration and biopsies revealed a lining of regular low cuboidal cells. Surgical removal was undertaken through a transperitoneal/retroperitoneal approach and pathology was consistent with a prostatic multilocular cystadenoma. Further studies are needed to characterize and classify cystic lesions of the prostate.
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