Chronic myeloid leukemia (CML) is one of the myeloproliferative disorders accounting for 15-20% of adult leukemia. Priapism is a rare initial presentation of CML occurring in 1-2% of cases. It is a urologic emergency requiring urgent multidisciplinary management as delay in initiating treatment may lead to erectile dysfunction. Combined treatment modalities are usually employed in its management including surgery, chemotherapy, therapeutic leukapheresis, and local intracavernous therapy. We report an 18-year-old male who presented with a 12-day history of priapism. Further evaluation with peripheral blood film revealed CML in chronic phase with ischemic prolonged priapism. At presentation patient refused consent for surgical intervention and subsequently had cytoreductive therapy with oral hydroxyurea. He achieved complete detumescence after 4 weeks of cytoreduction and by the 5th month during follow-up, he developed erectile dysfunction.