Hazelbag H M, Taminiau A H, Fleuren G J, Hogendoorn P C
Department of Pathology, University of Leiden, The Netherlands.
J Bone Joint Surg Am. 1994 Oct;76(10):1482-99. doi: 10.2106/00004623-199410000-00008.
The records of thirty-two patients who had had an adamantinoma of the long bones were examined to investigate the relationship between the clinical presentation, the histological subtype, and the method of treatment, and the clinical result. All histological patterns of differentiation that are characteristic of adamantinoma were observed, including the basaloid, spindle-cell, tubular, squamous, and osteofibrous dysplasia-like subtypes. Follow-up data were available for twenty-eight (88 per cent) of the thirty-two patients. These patients were followed for a mean duration of 122 months (range, eleven months to twenty-nine years and two months). Nine patients (32 per cent), all of whom had been managed with an intralesional or marginal procedure, had a local recurrence of the tumor after a mean disease developed in three of the nine patients. In five other patients, metastasis developed without having been preceded by a local recurrence. Thus, the over-all rate of metastasis was 29 per cent (eight patients). The mean duration of survival for the patients who had metastasis was twelve years and eight months. Statistical analysis of various clinicopathological variables revealed intralesional or marginal excision to be the most significant risk factor for a local recurrence or metastasis (p < 0.001). Two patients who had had a presumed osteofibrous dysplasia-like adamantinoma, which contained few isolated keratin-positive epithelial cells within the stroma at the time of presentation, had a full-blown adamantinoma at the time of the local recurrence. Although the clinical course that was observed may be the result of a sampling error, it poses questions as to the regressive nature of osteofibrous dysplasia-like adamantinoma. On the basis of our findings and the data in the literature, we believe that an osteofibrous dysplasia-like adamantinoma may be a precursor lesion of the classic type of adamantinoma.
对32例长骨造釉细胞瘤患者的记录进行了检查，以研究临床表现、组织学亚型、治疗方法与临床结果之间的关系。观察到了造釉细胞瘤所有具有特征性的分化组织学模式，包括基底样、梭形细胞、管状、鳞状和骨纤维发育不良样亚型。32例患者中有28例（88%）有随访数据。这些患者的平均随访时间为122个月（范围为11个月至29年零2个月）。9例患者（32%）均采用病损内或边缘性手术治疗，平均发病后肿瘤局部复发，其中3例患者出现远处转移。另外5例患者在未发生局部复发的情况下出现转移。因此，总的转移率为29%（8例患者）。发生转移的患者的平均生存时间为12年零8个月。对各种临床病理变量的统计分析显示，病损内或边缘性切除是局部复发或转移的最显著危险因素（p<0.001）。2例曾被推测为骨纤维发育不良样造釉细胞瘤的患者，初诊时间质内仅有少量孤立的角蛋白阳性上皮细胞，局部复发时发展为典型的造釉细胞瘤。尽管观察到的临床过程可能是抽样误差的结果，但它对骨纤维发育不良样造釉细胞瘤的退行性本质提出了疑问。根据我们的研究结果和文献数据，我们认为骨纤维发育不良样造釉细胞瘤可能是经典型造釉细胞瘤的前驱病变。
