Tung Nguyen, Melissa Tjota, Peng Wang, Antic Tatjana
{"title":"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。","authors":"Tung Nguyen, Melissa Tjota, Peng Wang, Antic Tatjana","doi":"10.1177/10668969251326254","DOIUrl":null,"url":null,"abstract":"<p><p>Secondary loss of INI1 immunohistochemical staining has been observed in various types of renal cell carcinoma (RCC), including <i>TFE3</i>-rearranged, fumarate hydratase-deficient RCC, and collecting duct carcinoma. We report the first tumor of secondary INI1 loss with <i>SMARCB1</i> gene alteration in hereditary succinate dehydrogenase (SDH)-deficient RCC that presented in a 39-year-old woman with a germline <i>SDHB</i> 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 <i>SDHB</i> variant (p.C191Y, VAF 77%) and an <i>SMARCB1</i> variant (p.P146Mfs33*, VAF 46%).</p>","PeriodicalId":14416,"journal":{"name":"International Journal of Surgical Pathology","volume":" ","pages":"10668969251326254"},"PeriodicalIF":0.9000,"publicationDate":"2025-03-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"A Case Report of INI1 Loss and <i>SMARCB1</i> Mutation in Hereditary Succinate Dehydrogenase-Deficient Renal Cell Carcinoma.\",\"authors\":\"Tung Nguyen, Melissa Tjota, Peng Wang, Antic Tatjana\",\"doi\":\"10.1177/10668969251326254\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Secondary loss of INI1 immunohistochemical staining has been observed in various types of renal cell carcinoma (RCC), including <i>TFE3</i>-rearranged, fumarate hydratase-deficient RCC, and collecting duct carcinoma. We report the first tumor of secondary INI1 loss with <i>SMARCB1</i> gene alteration in hereditary succinate dehydrogenase (SDH)-deficient RCC that presented in a 39-year-old woman with a germline <i>SDHB</i> 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 <i>SDHB</i> variant (p.C191Y, VAF 77%) and an <i>SMARCB1</i> variant (p.P146Mfs33*, VAF 46%).</p>\",\"PeriodicalId\":14416,\"journal\":{\"name\":\"International Journal of Surgical Pathology\",\"volume\":\" \",\"pages\":\"10668969251326254\"},\"PeriodicalIF\":0.9000,\"publicationDate\":\"2025-03-17\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"International Journal of Surgical Pathology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1177/10668969251326254\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"PATHOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"International Journal of Surgical Pathology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1177/10668969251326254","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"PATHOLOGY","Score":null,"Total":0}
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%).
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).