The authors present an unusual case of intramedullary arachnoid cyst diagnosed in a patient after the lesion was resected. A wide decompressive surgery was performed and the lesion removed. Histopathological findings were consistent with the diagnosis of arachnoid cyst. Postoperatively the patient exhibited marked improvement in neurological status. To the best of the authors' knowledge, there is no case report of intramedullary arachnoid cyst reported in the literature. With the advent of newer neuroimaging modalities such as magnetic resonance imaging the number of cases of intramedullary arachnoid cysts encountered in the future may increase.
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