Cyclosporine A (CsA) has assumed a central role in the prophylaxis of
acute graft-versus-host disease after allogenic bone marrow transplant
ation; however, its potential neurotoxicity can manifest in 10-35% of
patients. In most cases, neurotoxicity is transient, but fatal cases h
ave been described. We report a case of acute upbeating nystagmus appe
aring subsequent to the hematic peak of CsA in a patient undergoing pr
ophylactic treatment for acute graft-versus-host disease after bone ma
rrow transplantation for chronic myeloid leukemia. Computed tomography
and magnetic resonance imaging revealed no abnormalities. Although re
duced, the upbeating nystagmus persisted over time. To our knowledge,
this is the first case in which upbeating nystagmus manifests as a pos
sible consequence of CsA therapy.