多灶性血管肿瘤与 EWSR1::NFATC2 基因融合并发展为上皮样血管肉瘤--病例报告。

IF 3 3区 医学 Q1 PATHOLOGY Virchows Archiv Pub Date : 2024-12-01 Epub Date: 2024-11-04 DOI:10.1007/s00428-024-03962-x
Jože Pižem, Emanuela Boštjančič, Andrej Zupan, Vladka Salapura, Blaž Mavčič, Ana Blatnik, Olga Blatnik, Mojca Unk, Izidor Kern, Miha Švarc, Alenka Matjašič
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引用次数: 0

摘要

NFATC1/2融合的一类独特的血管肿瘤,累及骨骼和软组织,常表现局灶性上皮样形态,内皮细胞的可变异型性,主要是血管形成,在某些情况下,局灶性实体生长。虽然它们可能表现出侵略性的局部生长,并可能在局部复发,但恶性行为尚未被记录在案。我们报告一例35岁女性多发性血管肿瘤伴EWSR1::NFATC2融合,累及肺、多块骨(椎骨、股骨、胫骨、骨盆),并可能累及肝脏。骨病变局部侵袭,手术治疗后复发。首次表现后9年,进展为上皮样血管肉瘤。患者在诊断为上皮样血管肉瘤并大面积累及肺和肝脏(转移)后3个月死亡。除了EWSR1::NFATC2融合外,在血管肉瘤中发现了激活的PIK3CA基因突变,但在先前诊断的骨肿瘤中没有发现。据我们所知,这是首例血管肿瘤恶性进展并伴有NFATC1/2融合并累及内脏(肺)的病例。
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Multifocal vascular neoplasm with an EWSR1::NFATC2 gene fusion and progression to epithelioid angiosarcoma - a case report.

There is an emerging group of distinct vascular neoplasms with NFATC1/2 fusions, involving bones and soft tissues and often displaying focal epithelioid morphology, variable atypia of endothelial cells, predominantly vasoformative and in some cases focal solid growth. Although they may show aggressive local growth and may recur locally, malignant behaviour has not been documented. We present a case of a 35-year-old woman with multiple vascular neoplasms with a EWSR1::NFATC2 fusion involving the lungs, multiple bones (vertebra, femurs, tibia, pelvis) and probably the liver. The bone lesions were locally aggressive and recurred after surgical treatment. Nine years after the first manifestation, there was progression to an epithelioid angiosarcoma. The patient died 3 months after the diagnosis of epithelioid angiosarcoma with massive lung and liver involvement(metastases). In addition to the EWSR1::NFATC2 fusion, an activating PIK3CA gene mutation was identified in the angiosarcoma but not in the previously diagnosed bone tumours. To the best of our knowledge, this is the first documentation of malignant progression of a vascular neoplasm with NFATC1/2 fusion as well as visceral (lung) involvement.

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来源期刊
Virchows Archiv
Virchows Archiv 医学-病理学
CiteScore
7.40
自引率
2.90%
发文量
204
审稿时长
4-8 weeks
期刊介绍: Manuscripts of original studies reinforcing the evidence base of modern diagnostic pathology, using immunocytochemical, molecular and ultrastructural techniques, will be welcomed. In addition, papers on critical evaluation of diagnostic criteria but also broadsheets and guidelines with a solid evidence base will be considered. Consideration will also be given to reports of work in other fields relevant to the understanding of human pathology as well as manuscripts on the application of new methods and techniques in pathology. Submission of purely experimental articles is discouraged but manuscripts on experimental work applicable to diagnostic pathology are welcomed. Biomarker studies are welcomed but need to abide by strict rules (e.g. REMARK) of adequate sample size and relevant marker choice. Single marker studies on limited patient series without validated application will as a rule not be considered. Case reports will only be considered when they provide substantial new information with an impact on understanding disease or diagnostic practice.
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