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Epstein-Barr virus (EBV) associated central nervous system (CNS) infection is a rare disease. We report an atypical manifestation of EBV encephalitis initially presenting with a tumor-like lesion of the optic tract in an immunocompromised patient 8 years after a combined kidney and pancreas transplantation had been performed. Polymerase chain reaction (PCR) in the cerebrospinal fluid (CSF) and antibody testing confirmed the diagnosis of EBV encephalitis, most likely as a consequence of a reactivated persistent EBV infection.

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