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初次手术后41年发生的骨巨细胞瘤晚期恶性转化

Late malignant transformation of giant cell tumor of bone 41 years after primary surgery.

作者信息

Kadowaki Masaru, Yamamoto Soichiro, Uchio Yuji

机构信息

Department of Orthopaedic Surgery, Shimane University School of Medicine, Izumo-shi, Shimane, Japan.

出版信息

Orthopedics. 2012 Oct;35(10):e1566-70. doi: 10.3928/01477447-20120919-32.

DOI:10.3928/01477447-20120919-32
PMID:23027500
Abstract

Giant cell tumor of bone is an uncommon benign tumor that frequently recurs locally. Spontaneous malignant transformation of conventional giant cell tumor of bone is rare and usually occurs with irradiation.This article describes a case of malignant transformation of a giant cell tumor 41 years after initial curettage and subsequent resection. A 68-year-old man presented with a 6-month history of left hip pain. He had been diagnosed 41 years previously with giant cell tumor in the left femoral neck treated by simple curettage and bone grafting, followed by resection of the femoral head 1 year later for local recurrence. On presentation, radiographs revealed a destructive lesion in the left proximal femur. Incisional biopsy revealed recurrence of giant cell tumor with suspected malignant transformation. The patient underwent en bloc resection of the proximal femur with adequately wide margins and reconstruction of the hip joint with a prosthesis. Pathological findings showed malignant transformation of a giant cell tumor to osteosarcoma and leiomyosarcoma. No recurrence or metastasis developed during 2-year follow-up. Benign local recurrences usually arise in the first 3 postoperative years, whereas malignant transformation tends to take longer than 3 years. To the authors' knowledge, the 41-year interval from primary surgery to diagnosis of malignancy for the current patient is the longest interval reported among cases in which patients received no radiation therapy.

摘要

骨巨细胞瘤是一种罕见的良性肿瘤,常发生局部复发。传统骨巨细胞瘤的自发恶性转化罕见,通常与放疗有关。本文描述了一例骨巨细胞瘤在初次刮除及后续切除41年后发生恶性转化的病例。一名68岁男性,有6个月的左髋部疼痛病史。他在41年前被诊断为左股骨颈骨巨细胞瘤,接受了单纯刮除和骨移植治疗,1年后因局部复发进行了股骨头切除。就诊时,X线片显示左股骨近端有一破坏性病变。切开活检显示骨巨细胞瘤复发并疑似恶性转化。患者接受了近端股骨整块切除,切缘足够宽,并使用假体重建髋关节。病理结果显示骨巨细胞瘤恶变为骨肉瘤和平滑肌肉瘤。在2年的随访中未出现复发或转移。良性局部复发通常发生在术后前3年,而恶性转化往往需要3年以上时间。据作者所知,该患者从初次手术到诊断为恶性肿瘤间隔41年,是未接受放疗的病例中报道的最长间隔时间。

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