Sickle cell disease is a chronic congenital hemoglobinopathy with some major forms having high morbidity and mortality. The occurrence of chronic myeloid leukemia type malignant hematopathy in this area is rare but not excluded.

This association has drawn our attention to its clinical impact and the challenges of organizing care.

To this end, we report a case of chronic myeloid leukaemia on homozygous SSFA2 major sickle cell disease, diagnosed in a 35-year-old patient. He was treated with Imatinib Mesylate for CML and transfusional exchanges for sickle cell disease. Despite a toxicity noted at the beginning of treatment, the overall evolution thereafter was good with normalization of the blood count and stabilization of the clinical condition.