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Primary sclerosing epithelioid fibrosarcoma of bone: analysis of a series.

骨原发硬化性上皮样纤维肉瘤:系列病例分析

Wojcik JB,Bellizzi AM,Dal Cin P,Bredella MA,Fletcher CD,Hornicek FJ,Deshpande V,Hornick JL,Nielsen GP

Abstract

Sclerosing epithelioid fibrosarcoma (SEF) is a rare, aggressive malignant neoplasm characterized by small nests and linear arrays of epithelioid cells embedded in a dense collagenous matrix. Very few primary SEFs of bone have been reported. Recognition is critical, as the dense extracellular collagenous matrix can be interpreted as osteoid, leading to misdiagnosis as-osteosarcoma. MUC4 and SATB2 are 2 recently characterized immunohistochemical markers for SEF and osteosarcoma, respectively. In reports to date, osteosarcomas are positive for SATB2 and negative for MUC4, whereas soft tissue SEFs have shown the opposite immunohistochemical profile (SATB2-/MUC4+). The purpose of this study was to characterize the clinicopathologic and immunohistochemical features of 8 primary SEFs of bone. The patients presented at a wide range of ages (25 to 73 y; median 52 y). Tumors mostly involved long bones of the extremities, with 3 cases involving the femur, 2 involving the ulna, and 1 involving the humerus. Other sites of involvement included the second rib (1) and the C6 vertebra (1). Follow-up information was available for 7 patients, 3 of whom developed metastases within 2 years of diagnosis. The other 4 patients were free of local recurrence or metastases at 1, 5, 12, and >84 months of follow-up, respectively. Radiographically, the tumors were predominantly lytic and poorly marginated. Histologically, 6 tumors showed pure SEF morphology, and 2 showed hybrid SEF/low-grade fibromyxoid sarcoma morphology. Focal dystrophic mineralization was seen in 1 case but was limited to areas of necrosis. None of the tumors showed the lace-like pattern of mineralization typical of osteosarcoma. The majority (6/8) of the tumors strongly expressed MUC4. SATB2 was negative in all but 1 case, which showed variable weak to moderate staining in ∼50% of nuclei. In general, the combination of morphology, MUC4 expression, and the absence of SATB2 expression was highly useful in arriving at the correct diagnosis.

摘要

硬化性上皮样纤维肉瘤(SEF)是一种罕见的侵袭性恶性肿瘤,以埋陷于致密胶原基质中的小巢状和线性排列上皮样细胞为特征。极其少见的骨原发性SEFs已见报道。识别该病变至关重要,因致密细胞外胶原基质可被解读为骨样基质,导致误诊为骨肉瘤。最近发现MUC4和SATB2分别是SEF和骨肉瘤特征性的免疫组织化学标记物。到目前为止的报道中,骨肉瘤SATB2阳性而MUC4阴性,而软组织SEFs呈相反的免疫组织化学表达(SATB2-/MUC4+)。本研究的目的是描述8例骨原发性SEFs的临床病理学和免疫组织化学特征。患者年龄范围宽(25-73岁;中位52岁)。肿瘤主要累犯四肢长骨,3例累犯股骨,2例累犯尺骨,1例累犯肱骨。其他受累部位包括第二肋骨(1)和C6椎骨(1)。7名患者有随访信息,其中3例诊断2年内发生转移。其他4名患者分别在随访1、5、12和多于84个月时无局部复发或转移。放射学检查发现肿瘤显著溶骨性改变,边界不清。组织学,6例肿瘤显示纯SEF形态学,2例显示混合性SEF/低级别纤维粘液样肉瘤形态。1例可见灶性营养不良性骨化但限于坏死区域。没有肿瘤出现骨肉瘤典型的带样骨化模式。大多数(6/8)肿瘤高表达MUC4. 除1例肿瘤约50%细胞核呈SATB2弱到中度染色外,其余全部呈SATB2阴性。总之,结合形态学,MUC4表达和SATB2缺失有助于得出正确的诊断。
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