Acute encephalopathy is a recognized toxic effect of Cyclosporin A (CsA) in organ transplantation recipients. A 16-year-old girl presented with acute encephalopathy 2 weeks after CsA and methylprednisolone medication for idiopathic uveitis. Magnetic resonance imaging showed cortical and white matter occipital changes, which were not visible 2 months later. With expanding indication for CsA use, an increasing number of neurotoxic cases are to be expected.