The case of a 63-year-old woman is presented who developed 18 years after undergoing allogenic bone marrow transplantation for chronic myeloid leukaemia a granulocytic sarcoma localised in the presacral region. Complete radiologic remission was obtained with the administration of imatinib. She developed a metachron granulocytic sarcoma in the right frontal region associated to leptomeningeal infiltration. Complete clinical and near complete radiological remission was obtained with the administration of nilotinib. The patient was finally lost of progression of the frontal and meningeal localisations 45 months after the presentation of the first presacral lesion. Throughout the follow-up no sign of systemic leukaemia was detected.