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An 18-year-old Thai male presented with weight loss and urinary retention. Physical examination revealed an enlarged prostate gland. Serum prostate specific antigen (PSA) level was 0.3ng/ml. Histologic section demonstrated a small round cell tumor with intracytoplasmic glycogen. Immunohistochemical and ultrastructural studies were compatible with Ewing's sarcoma. Since no other osseous or extraosseous lesions of the same tumor were found in this patient, we concluded that this was a case of primary extraskeletal Ewing's sarcoma of the prostate gland. Case follow-up after one year revealed a good response to chemotherapy.
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