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Pleomorphic xanthoastrocytoma is a rare, cortically based, partially cystic astroglial tumour with a strongly enhancing solid mural nodule. We report an 11-year-old boy with a pleomorphic xanthoastrocytoma in the contralateral hemisphere to that affected by Sturge-Weber syndrome. This unique case supports the association between Sturge-Weber syndrome and astrocytomas. Patients with Sturge-Weber syndrome who experience unexpected neurological symptoms should be evaluated with MRI.