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30 例遗传性肾细胞癌综合征患者的 60 个遗传性肾肿瘤的临床和形态学复习:来自当代单一机构系列的经验教训。

Clinical and morphologic review of 60 hereditary renal tumors from 30 hereditary renal cell carcinoma syndrome patients: lessons from a contemporary single institution series.

机构信息

Department of Pathology, University of Michigan Medical School, Ann Arbor, MI, USA.

Michigan Center for Translational Pathology, Ann Arbor, MI, USA.

出版信息

Med Oncol. 2019 Jul 22;36(9):74. doi: 10.1007/s12032-019-1297-6.

Abstract

Hereditary renal cell carcinoma syndromes (HRCCS) are characterized by the presence of pathogenic germline variants that predispose patients to renal cell carcinomas as well as additional extra-renal manifestations. The importance of identifying HRCCS patients cannot be overemphasized, as patients and their families can begin surveillance for syndrome-associated manifestations once identified. The present study is a retrospective clinical and morphologic review of 60 hereditary renal tumors from 30 HRCCS patients treated at our institution with either Von Hippel-Lindau disease (VHL), Birt-Hogg-Dubé syndrome (BHD), tuberous sclerosis complex (TSC), hereditary leiomyomatosis and renal cell cancer syndrome, or succinate dehydrogenase (SDH) deficiency syndrome. Hereditary renal cell carcinoma syndromes kidney tumors often demonstrate specific morphologic features, characteristic background changes in renal parenchyma, and extra-renal manifestations, which, when recognized by the pathologist, can trigger genetic testing referral for specific familial cancer syndromes. Our study demonstrates the majority of tumors were consistent with the anticipated clinicopathologic profile of renal tumors found within HRCCS patients, although we found some unique characteristics within this cohort including a case of clear cell papillary renal cell carcinoma within a VHL patient, and a unique renal tumor with tubulopapillary features present in a patient with a germline SDHD mutation. Additionally, although the literature reports the presence of epithelioid angiomyolipoma (AML) as a common occurrence in TSC patients, our cohort of 3 patients with AMLs demonstrated only classic features. The findings we describe facilitate pathologist-based recognition of HRCCS and can prompt genetic evaluation for relevant patients.

摘要

遗传性肾细胞癌综合征 (HRCCS) 的特征是存在致病性种系变异,使患者易患肾细胞癌以及其他肾脏外表现。识别 HRCCS 患者的重要性怎么强调都不为过,因为一旦确定,患者及其家属可以开始对综合征相关表现进行监测。本研究回顾性分析了我院 30 例 HRCCS 患者的 60 例遗传性肾肿瘤,这些患者分别患有 von Hippel-Lindau 病 (VHL)、Birt-Hogg-Dubé 综合征 (BHD)、结节性硬化症、遗传性平滑肌瘤和肾细胞癌综合征或琥珀酸脱氢酶 (SDH) 缺乏综合征。遗传性肾细胞癌综合征的肾肿瘤常表现出特定的形态学特征、肾实质的特征性背景改变和肾脏外表现,病理学家识别这些特征后,可提示进行特定家族性癌症综合征的基因检测。我们的研究表明,大多数肿瘤与 HRCCS 患者肾肿瘤的预期临床病理特征一致,尽管我们在该队列中发现了一些独特的特征,包括 VHL 患者中存在透明细胞乳头状肾细胞癌,以及携带种系 SDHD 突变的患者中存在具有管状乳头状特征的独特肾肿瘤。此外,尽管文献报道上皮样血管平滑肌脂肪瘤 (AML) 是 TSC 患者的常见表现,但我们的 AML 患者队列中仅表现出经典特征。我们描述的这些发现有助于病理学家识别 HRCCS,并可为相关患者提供遗传评估。

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