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The evidence of CNS involvement in Ewing's sarcoma is uncommon and very often due to a direct extension of the tumor to extradural space and meninges. We report a case of a 14-year old girl with a primary ischio-pubic localization of Ewing's sarcoma. Body CT scan showed a bone lesion involving the left fronto-temporal region of the skull without affecting the brain. After chemotherapy radiological findings showed complete recovery. Ten months after treatment, the patient had neurological symptoms. Cerebral CT scan evidenced a large tumor mass localized in the right temporal lobe. Following radiotherapy the tumor mass was no longer visible. This case shows that late intraparenchymal CNS metastasis occurs independently from the previous skull lesion. Therefore these data, even through based on a single observation, lead to believe that in some cases prophylactic chemotherapy and radiotherapy of CNS may not be necessary.
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