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MEIS1::NCOA1 肾原发性梭形细胞肉瘤:7例独特临床病理实体的报告

MEIS1::NCOA1 Primitive Spindle Cell Sarcoma of the Kidney : Report of 7 Cases of a Distinctive Clinicopathologic Entity.

作者信息

Argani Pedram, Wangsiricharoen Sintawat, Tretiakova Maria, Liu Yajuan J, Falzarano Sara M, Collins Katrina, Brimo Fadi, Gross John M, Baraban Ezra, Matoso Andres, Wakeman Kristina, Corless Christopher, Neff Tanaya, Smith Benjamin F, Abdel Satir Ali, Agaimy Abbas, Antonescu Cristina R, Charville Gregory W, Sangoi Ankur R

机构信息

Departments of Pathology and Oncology, Johns Hopkins University School of Medicine, Baltimore, MD.

Department of Pathology and Laboratory Medicine, Oregon Health & Science University, Portland, OR.

出版信息

Am J Surg Pathol. 2025 Jun 1;49(6):620-632. doi: 10.1097/PAS.0000000000002386. Epub 2025 Mar 31.

Abstract

Primitive sarcomas harboring the MEIS1::NCOA2 gene fusion were originally described in the kidney in 2018, and subsequently reported in other organs. These variably cellular neoplasms feature monomorphic primitive plump spindle cells forming nodules and whorls in addition to nondescript fascicular, solid, and storiform patterns. They lack skeletal muscle differentiation in contrast to the primarily intraosseous rhabdomyosarcomas that harbor the same gene fusion. We describe 7 new primary primitive renal sarcomas with MEIS1::NCOA1 gene fusions. Although their morphology overlaps with that described in MEIS1::NCOA2 renal sarcoma, 3 of the 7 cases contained adipose tissue. The majority had intimately admixed entrapped cystic epithelial elements and demonstrated patchy immunoreactivity for estrogen receptor and nuclear labeling for WT1 protein, leading to the differential diagnosis of malignant mixed epithelial stromal tumor (MEST) in 4 cases and metanephric stromal tumor in one. The neoplasms demonstrate a broad spectrum of clinicopathologic features ranging from a bland low-grade neoplasm that metastasized 9 years after diagnosis to a high-grade sarcoma with multiple recurrences, ultimately leading to patient death in under 1 year. In summary, MEIS1::NCOA1 primitive sarcomas overlap with the previously described MEIS1::NCOA2 primitive renal sarcomas and represent a distinctive renal neoplasm that can be mistaken for malignant MEST. Grade ranges from low to high but even low-grade neoplasms require long-term clinical follow-up.

摘要

2018年首次在肾脏中发现携带MEIS1::NCOA2基因融合的原始肉瘤,随后在其他器官中也有报道。这些肿瘤细胞成分各异,除了有形态不典型的束状、实性和席纹状结构外,还具有单形性原始丰满梭形细胞形成结节和漩涡状结构的特征。与同样携带该基因融合的主要发生于骨内的横纹肌肉瘤不同,它们缺乏骨骼肌分化。我们描述了7例新的原发性携带MEIS1::NCOA1基因融合的原始肾肉瘤。尽管它们的形态与MEIS1::NCOA2肾肉瘤中描述的相似,但7例中有3例含有脂肪组织。大多数肿瘤紧密混合有陷入的囊性上皮成分,并对雌激素受体呈局灶性免疫反应,WT1蛋白呈核标记阳性,导致4例被误诊为恶性混合上皮间质瘤(MEST),1例被误诊为后肾间质瘤。这些肿瘤表现出广泛的临床病理特征,从诊断后9年发生转移的低级别温和肿瘤到多次复发的高级别肉瘤,最终在不到1年的时间内导致患者死亡。总之,MEIS1::NCOA1原始肉瘤与先前描述的MEIS1::NCOA2原始肾肉瘤有重叠,代表一种独特的肾肿瘤,可被误诊为恶性MEST。分级从低到高,但即使是低级别肿瘤也需要长期临床随访。

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