A 21-year-old Caucasian man received an allogeneic marrow transplant (BMT) from his HLA-identical brother because of myelodysplastic syndrome. He remained red blood cell (RBC) transfusion dependent with persistent antibodies against the donor's RBC. Six months following BMT the patient suddenly developed a severe akinetic syndrome with gait disturbance and frequent falls and bilateral symmetrical lesions in basal ganglia. Concomitantly, micrococcus species septicemia from an infected Hickman catheter developed. Despite antimicrobial therapy and withdrawal of cyclosporin A, neurologic abnormalities persisted and were unresponsive to various therapies. Ischemic damage due to a vascular event during severe infection could be the most probable reason for the lesions seen in our patient, although infectious or toxic complications cannot be ruled out.