Priapism or prolonged erection of the penis is extremely rare in pediatric population, and occurs mostly in hemoglobinopathies, hypercoagulable states, in cancer and spinal injuries. There are two types of priapism: high and low blood flow, and their differentiation is based on the clinical picture and the study of blood gas analysis of blood aspirated from the corpus cavernosum. Ischemic priapism is a condition requiring urgent intervention and currently the gold standard adopts the aspiration of the corpora cavernosa rinsing with saline and drug administration - (sympathomimetics and alpha-adrenergic agonists). In this paper we present a case of priapism in a 9-year-old boy with acute lymphoblastic leukemia (ALL).