An aggressively poor course of primary orbital pleomorphic liposarcoma is presented. A 59-year-old man had suffered from multiple local recurrences and died 1 year after diagnosis of orbital pleomorphic liposarcoma. The tumor invaded into the brain only 10 months after exenteration. The patient had a history of partial tumor resection via anterior orbitotomy before total exenteration. It was assume that such an unusual poor course might be due to histologic findings, which revealed a high-grade pleomorphic liposarcoma with a high mitotic count (25/10 high-power field) or previous-done anterior orbitotomy, which was able to seed the tumor cells.
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