Priapism in Chronic Myelogenous Leukemia(CML) is an unusual presentation but some cases are diagnosed due to complain of priapism. Priapism may be due to leukostasis associated with greatly exaggerated blood leukocyte count as low flow (ischemic) or high flow (non ischemic) type. Treatment option includes- aspiration, sympathomimetic agents, leukapheresis, cytotoxic therapy and shunt surgery. Study case was a 11-year-old boy presented with priapism and fullness in abdomen. Careful physical examination and blood investigations revealed CML. We treated conservatively and priapism resolved in 24 hours.