Priapism is an unusual andrological emergency that requires urgent intervention. It is characterized by prolonged penile erection for more than 4h not related to sexual excitation. It is classified into three main types: venoocclusive (ischemic), arterial (nonischemic) and stuttering priapism. Many causes can predispose to ischemic priapism including thalassemias, sickle cell anemia, hypercoagulable status, neoplastic syndromes, pelviabdominal tumors and use of some drugs and medications. However, in the majority of cases of ischaemic priapism no cause could be identified. Chronic myeloid leukemia (CML) is a chronic myeloprolifertative tumor that is characterized by a variety of vague clinical manifestations. Priapism as a complication of CML is a rare clinical presentation. It may cause priapism as a result of hypercoagulability due to hyperleukocytosis. Here, in this report, the case of a known CML male patient who presented with venoocclusive priapism for about 16 h after abrupt discontinuation of his cytoreductive therapy is described. Thus, all medical staff, either (General practitioners) GPs or specialized in different fields of medicine, should be aware of priapism as an emergency and that early diagnosis and multidisciplinary management minimize the inevitable damage of the erectile corpora cavernosa as well as improving the outcome