Priapism is a rare disease. It is an emergency condition with a poor prognosis, and the risk of impotence is 50% despite appropriate management. Though about 20% cases of all priapism are related to hematological disorders, the incidence of priapism in adult leukemic patients is only about 1-5 percent. The incidence in pediatric leukemia patient is even rarer. Here we present a case of priapism in a 14-year-old apparently healthy boy who found to have chronic myeloid leukemia on subsequent investigations.