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Introduction The management of recurrent ischemic priapism is unclear in contemporary practice. Yet, if left untreated, the condition may evolve into an acute ischemic priapism and in some cases result in erectile dysfunction. This report documents the results of successful management of recurrent ischemic priapism using cyproterone acetate in a 30-year-old Saudi man with sickle cell anemia as a comorbidity. Case presentation A 30-year-old Saudi man denoted visited the emergency room with a painful erection which had lasted for more than four hours. The patient has sickle cell anemia and a family history of sickle cell disease. He is married and has two children. His first priapism case occurred when he was 7 years old. At the age of 15, the condition recurred, and the patient’s doctor prescribed cyproterone acetate 50 mg twice daily for 5 days. The doctor had told him that whenever he was experiencing priapism, he should adhere to this regimen for 5 days. The doctor could not find any guidelines for the prescription of cyproterone acetate. Conclusion Priapism cases represent a significant challenge in therapeutic management because of the elevated risk of structural damage to the penis. The fact that there lacks a clinically approved standard approach to managing the condition make it difficult for physicians to effectively manage the condition. Management of the condition is further complicated by existence of comorbidities such as sickle cell anemia. This patient’s case demonstrates that cyproterone acetate prescription is a great preventative strategy that limits priapism recurrences.