Abstract We report the case of a patient who was initially presented with ischemic priapism to the emergency department. He was treated with adrenaline intracavernous injections and aspiration with irrigation of the corpora cavernosa and distal shunt. In the postoperative period, anemia, basophilia, eosinophilia, thrombocytosis and hyperleukocytosis were detected. The patient was subsequently diagnosed with chronic myeloid leukemia. Priapism is a rare manifestation of chronic myeloid leukemia (≤ 3%) and occurs mostly due to hyperleukocytosis, resulting in thrombus formation and corporal venous outflow obstruction. Priapism occurring in any setting is considered as a medical emergency that requires immediate local therapy because of resulting irreversible cell damage and fibrosis if not treated within the first 24–48 h.