The koro syndrome is a psychiatric disorder characterized by acute anxiety and a deep-seated fear of shrinkage of the penis and its ultimate retraction into the abdomen, which will cause death. Concurrence of the koro (genital retraction) syndrome with a pathological condition of the urogenital system has rarely been described. We report a case of koro associated with infertility. Within 3 weeks of treatment with haloperidol the classic symptoms of koro disappeared. To our knowledge this case represents the sixth report of the koro syndrome associated with urogenital pathology and the first report of its association with infertility.