Institute of Pathology, University Hospital, 91054 Erlangen, Germany.
Department of Pathology, Faculty of Medicine in Pilsen, Charles University, 30460 Plzen, Prague; Biomedical Center, Faculty of Medicine in Pilsen, Charles University, 30460 Plzen, Czech Republic.
Hum Pathol. 2018 Jul;77:20-27. doi: 10.1016/j.humpath.2017.12.025. Epub 2018 Jan 5.
Dedifferentiated liposarcoma is one of the most common sarcoma types in adults with a predilection for the retroperitoneum. We have recently encountered 6 cases of DDL composed predominantly of rounded, rhabdoid or epithelioid cells mimicking rhabdoid melanoma, epithelioid rhabdomyosarcoma or undifferentiated carcinoma. Patients were 5 males and one female aged 64 to 81 years (median, 68). Tumors originated in the retroperitoneum (n=5; 3 in the psoas muscle) and deep soft tissue of the thigh (n=1). All 3 patients with follow-up died of metastatic disease within 4 to 8 months. Preoperative biopsy diagnoses never suggested dedifferentiated liposarcoma as a possibility; instead carcinoma, rhabdomyosarcoma and lymphoma were on top of suggestions. Five resected tumors were composed predominantly (70%-100%) of anaplastic rounded to oval rhabdoid cells with prominent central nucleoli and paranuclear rhabdoid inclusions. Bi- and multinucleation was a constant feature. The background stroma showed variable myxoid changes and minor mixed inflammatory cells. Two cases showed homologous dedifferentiation and another had sclerosing spindle cell nodule but a well-differentiated lipomatous component was not seen in any. One biopsied case showed solely monotonous small round blue cells with scattered rhabdoid cells. Immunohistochemistry showed expression of MDM2 (6/6), CDK4 (5/6), pancytokeratin AE/1AE3 (4/6) and diffusely desmin and myogenin (2/6). All cases showed high-level co-amplification of MDM2/CDK4 by in situ hybridization. The SWI/SNF complex components (SMARCB1, SMARCA2, SMARCA4, ARID1A and PBRM1) were intact in all cases. This highly aggressive liposarcoma variant needs to be distinguished from a variety of neoplasms including undifferentiated carcinoma, melanoma, lymphoma, rhabdomyosarcoma and others.
去分化脂肪肉瘤是成人中最常见的肉瘤类型之一,偏爱腹膜后。我们最近遇到了 6 例主要由圆形、横纹肌样或上皮样细胞组成的 DDL,这些细胞类似于横纹肌肉瘤、上皮样横纹肌肉瘤或未分化癌。患者为 5 名男性和 1 名女性,年龄 64 至 81 岁(中位数,68 岁)。肿瘤起源于腹膜后(n=5;3 例位于腰大肌)和大腿深部软组织(n=1)。所有 3 例有随访的患者均在 4 至 8 个月内死于转移性疾病。术前活检诊断从未提示去分化脂肪肉瘤的可能性;相反,癌、横纹肌肉瘤和淋巴瘤是最有可能的诊断。5 例切除的肿瘤主要由(70%-100%)多形性圆形至椭圆形横纹肌样细胞组成,具有明显的中央核仁及核周横纹肌样包涵体。双核和多核是一个恒定的特征。背景基质显示出不同程度的黏液样变化和少量混合性炎症细胞。2 例有同源性去分化,另 1 例有硬化性梭形细胞结节,但任何病例均未见明显的分化良好的脂肪成分。1 例活检病例仅表现为单调的小圆形蓝色细胞,伴有散在的横纹肌样细胞。免疫组化显示 MDM2(6/6)、CDK4(5/6)、广谱细胞角蛋白 AE/1AE3(4/6)和弥漫性 desmin 和 myogenin(2/6)表达。所有病例均通过原位杂交显示 MDM2/CDK4 高水平共扩增。SWI/SNF 复合物成分(SMARCB1、SMARCA2、SMARCA4、ARID1A 和 PBRM1)在所有病例中均完整。这种具有高度侵袭性的脂肪肉瘤变异需要与多种肿瘤相鉴别,包括未分化癌、黑色素瘤、淋巴瘤、横纹肌肉瘤等。