Schreiner Andrew, Daneshmand Siamak, Bayne Aaron, Countryman Gayle, Corless Christopher L, Troxell Megan L
Department of Pathology, Division of Urology & Renal Transplantation, Oregon Health & Science University, Portland, OR 97239, USA.
Int J Surg Pathol. 2010 Oct;18(5):409-18. doi: 10.1177/1066896909333510. Epub 2009 Apr 29.
Tuberous sclerosis complex results from mutations in 1 of 2 interacting gene products, hamartin or tuberin. The syndrome is characterized by hamartomas and neoplastic lesions, including angiomyolipomas of the kidney and other organs. Renal cell carcinoma (RCC) in tuberous sclerosis remains relatively poorly characterized because historical studies were confounded by the inclusion of epithelioid angiomyolipomas. The authors present a patient with tuberous sclerosis and bilateral renal lesions, including multiple minute angiomyolipomas, cortical cysts, and 4 separate RCCs of unclassified type. The carcinomas shared distinctive morphological features, including sheet-like, glandular, trabecular, or cystic architecture and abundant granular eosinophilic cytoplasm. By definition, the carcinomas were keratin positive and negative for HMB-45 and Melan-A. Detailed immunohistochemical analysis revealed heterogeneity among the cortical cysts and carcinomas. The histopathological features of these carcinomas illustrate characteristics of renal carcinoma that are probably related to genetic alterations of tuberous sclerosis.
结节性硬化症是由两种相互作用的基因产物(错构瘤蛋白或结节蛋白)之一发生突变引起的。该综合征的特征是错构瘤和肿瘤性病变,包括肾脏及其他器官的血管平滑肌脂肪瘤。结节性硬化症中的肾细胞癌(RCC)特征相对不明确,因为既往研究因纳入上皮样血管平滑肌脂肪瘤而受到混淆。作者报告了一名患有结节性硬化症和双侧肾脏病变的患者,包括多个微小血管平滑肌脂肪瘤、皮质囊肿以及4个未分类的独立肾细胞癌。这些癌具有独特的形态学特征,包括片状、腺管状、小梁状或囊性结构以及丰富的颗粒状嗜酸性细胞质。根据定义,这些癌角蛋白呈阳性,而HMB-45和Melan-A呈阴性。详细的免疫组化分析显示皮质囊肿和癌之间存在异质性。这些癌的组织病理学特征说明了可能与结节性硬化症基因改变相关的肾细胞癌特征。