Priapism is a rare clinical presentation of a patient with chronic myeloid leukaemia (CML). Herein, we present a young Nepalese man that presented to the emergency department with an acute and painful penile erection for two days. Clinically, he was pale and abdominal examination revealed hepatomegaly. Combined oncologic and initial urological intervention with carvernosal aspiration and intracavernosal phenylephrine failed to achieve detumescence. The patient underwent an emergency corporoglandular shunting eventually. In this case report, we discuss the management compared with previously reported cases.