Priapism is a urological emergency. The treatment for ischaemic priapism is usually cavernosal aspiration with or without cavernosal irrigation. Some patients may need surgical intervention -the various shunt procedures. We report a 21-year-old man with priapism secondary to chronic myeloid leukemia who needed a combined medical and surgical management. He underwent a spongiocavernosal shunt as well as cytoreductive chemotherapy to achieve complete detumescence. Therefore, cytoreductive chemotherapy is an adjunct in difficult cult to treat priapism associated with chronic myeloid leukemia.