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新型 EWSR1-SSX3 基因融合拓展了骨未分化肉瘤的分子谱。

Expanding the molecular landscape of undifferentiated sarcomas of bone with a novel EWSR1-SSX3 gene fusion.

机构信息

Department of Pathology, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain.

Department of Oncology, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain.

出版信息

Genes Chromosomes Cancer. 2024 Jan;63(1):e23215. doi: 10.1002/gcc.23215. Epub 2023 Dec 5.

Abstract

Undifferentiated sarcomas characterized by a primitive monomorphic round to spindle cell phenotype and often non-specific immunoprofile remain difficult to subclassify outside molecular analysis. The increased application of RNA sequencing in clinical practice led to significant advances and discoveries of novel gene fusions that furthered our understanding and refined classification of otherwise undifferentiated neoplasms. In this study, we report an undifferentiated round to spindle cell sarcoma arising in the femur of a 34-year-old female. The round to spindle tumor cells were arranged in short fascicles, with focal rosette formation, within a hyalinized stroma. The tumor immunoprofile included diffuse reactivity for CD99, SATB2, and TLE1 and patchy positivity for Cyclin D1, Keratin AE1/AE3, synaptophysin, and chromogranin. Other markers, such as EMA, SMA, desmin, S100, ERG, and WT1, were negative. Fluorescence in situ hybridization analysis for EWSR1 gene alterations showed a break-apart signal and targeted RNA sequencing revealed an EWSR1::SSX3 gene fusion. The patient received neoadjuvant chemotherapy followed by surgery and subsequently relapsed in less than a year with lung metastasis. Larger series are needed to determine if this fusion defines a novel subset of undifferentiated tumors or represents a genomic variant of already existing primitive round cell sarcoma categories, such as Ewing sarcoma or synovial sarcoma.

摘要

未分化的肉瘤表现为原始的单一形态圆形至梭形细胞表型,且免疫表型通常不具有特异性,在没有分子分析的情况下仍然难以进一步细分亚类。随着 RNA 测序在临床实践中的广泛应用,我们对新型基因融合的认识取得了显著的进展和突破,这些发现进一步加深了我们对未分化肿瘤的理解并对其进行了更精细的分类。在本研究中,我们报告了一例发生于 34 岁女性股骨的未分化圆形至梭形细胞肉瘤。圆形至梭形肿瘤细胞排列成短束状,在玻璃样基质中具有局灶性玫瑰花结形成。肿瘤免疫表型包括 CD99、SATB2 和 TLE1 的弥漫性反应性,以及 Cyclin D1、角蛋白 AE1/AE3、突触素和嗜铬粒蛋白的局灶性阳性。其他标志物,如 EMA、SMA、结蛋白、S100、ERG 和 WT1,则为阴性。EWSR1 基因改变的荧光原位杂交分析显示分离信号,靶向 RNA 测序显示存在 EWSR1::SSX3 基因融合。患者接受了新辅助化疗,随后进行了手术,不到一年后出现肺转移复发。需要更大的系列研究来确定这种融合是否定义了一种新的未分化肿瘤亚类,还是代表了已经存在的原始圆形细胞肉瘤类别的基因组变异,如尤文肉瘤或滑膜肉瘤。

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