BACKGROUND: Priapism is a rare complication seen in one to five percent of adult leukemic patients. The word 'Priapism' is related to Priapus, the Greek and Roman God of procreation whose symbol was an erect phallus.

CLINICAL PRESENTATION: The patient is a 22-year-old male with no known co-morbidities presenting with one month intermittent, unstimulated, painful penile erection with no other associated symptoms which resolves spontaneously, until nine hours prior to admission when symptoms recurred and persisted. Patient had no history of trauma and no drug intake.

PHYSICAL FINDINGS: Patient was awake, in pain and tachycardic. There was note of pallor and splenomegaly. The penis was erect, firm, swollen and tender with superficial venous engorgement. The rest of the physical examination was unremarkable.

LABORATORY WORK UP: Complete blood count showed anemia and leukocystosis. Peripheral blood smear revealed markedly increased white blood cells with predominance of mature and immature cells belonging to granulocytic series. There was splenomegaly on ultrasound. Genetic testing showed an abnormal male karyotype of 46 chromosomes including translocation (9;22).

TREATMENT: Corpora cavernosa aspiration was done. Terbutaline was given. Patient was started and maintained on hydroxyurea and presently enrolled in Imitanib study.

OUTCOME: There was resolution of priapism after the corpus cavernosa aspiration and initiation of hydroxyurea and the white blood cell count had decreased after initiation of hydroxyurea.