Priapism is a painful and prolonged erection occurring without any sexual stimulation and not resulting in ejaculation. It most often occurs in patients with sickle cell disease. We report here the case of acute recurrent priapism revealing inaugural mode of CML in a 22-year-old patient with no particular pathological history. The study of the onco-hematological karyotype revealed a karyotype with 46 chromosomes with a clonal chromosomal anomaly: The t (9; 22) “Philadelphia chromosome”. The evolution was favorable under imatinib.
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