遗传性琥珀酸脱氢酶缺陷肾细胞癌中INI1缺失和SMARCB1突变1例报告。

IF 0.9 4区 医学 Q4 PATHOLOGY
Tung Nguyen, Melissa Tjota, Peng Wang, Antic Tatjana
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引用次数: 0

摘要

在各种类型的肾细胞癌(RCC)中,包括tfe3重排、富马酸水合酶缺陷的RCC和收集管癌,都观察到INI1免疫组化染色的继发性丧失。我们报告了遗传性琥珀酸脱氢酶(SDH)缺陷RCC中继发性INI1缺失伴SMARCB1基因改变的第一例肿瘤,该肿瘤出现在一名患有种系SDHB突变的39岁女性中。肿瘤表现为棕褐色肿块伴局灶性出血,浸润肾窦和肾周脂肪组织。镜下可见不同的建筑模式,包括实性、管状、假乳头状和肉瘤样区伴局灶性坏死。间质硬化、透明化,伴骨化生。尽管SDHB缺失的RCC表现出非典型的细胞形态,但SDHB的免疫组化在整个过程中都缺失。INI1在低等级区域保留,而在高等级区域丢失。下一代测序鉴定出致病性SDHB变异(p.p c191y, VAF 77%)和SMARCB1变异(p.p p146mfs33 *, VAF 46%)。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A Case Report of INI1 Loss and SMARCB1 Mutation in Hereditary Succinate Dehydrogenase-Deficient Renal Cell Carcinoma.

Secondary loss of INI1 immunohistochemical staining has been observed in various types of renal cell carcinoma (RCC), including TFE3-rearranged, fumarate hydratase-deficient RCC, and collecting duct carcinoma. We report the first tumor of secondary INI1 loss with SMARCB1 gene alteration in hereditary succinate dehydrogenase (SDH)-deficient RCC that presented in a 39-year-old woman with a germline SDHB mutation. The tumor appeared as a tan-brown mass with focal hemorrhage, infiltrating the renal sinus and perinephric adipose tissue. Microscopically, it showed diverse architectural patterns, including solid, tubular, pseudopapillary, and sarcomatoid areas with focal necrosis. The stroma showed sclerotic and hyalinized changes with osseous metaplasia. Immunohistochemistry for SDHB was lost throughout, despite the tumor showing nonclassic cytomorphology for SDH-deficient RCC. INI1 retention was noted in low-grade areas, while it was lost in high-grade regions. Next-generation sequencing identified a pathogenic SDHB variant (p.C191Y, VAF 77%) and an SMARCB1 variant (p.P146Mfs33*, VAF 46%).

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来源期刊
CiteScore
2.10
自引率
0.00%
发文量
198
审稿时长
1 months
期刊介绍: International Journal of Surgical Pathology (IJSP) is a peer-reviewed journal published eight times a year, which offers original research and observations covering all major organ systems, timely reviews of new techniques and procedures, discussions of controversies in surgical pathology, case reports, and images in pathology. This journal is a member of the Committee on Publication Ethics (COPE).
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