Martignoni Guido, Pea Maurizio, Zampini Claudia, Brunelli Matteo, Segala Diego, Zamboni Giuseppe, Bonetti Franco
Department of Pathology and Diagnostics, University of Verona, P.Le L.A. Scuro n. 10, Verona 37134, Italy; Department of Pathology, Pederzoli Hospital, Peschiera, Verona, Italy.
Department of Pathology, Orlandi Hospital, Bussolengo, Verona, Italy.
Semin Diagn Pathol. 2015 Mar;32(2):140-59. doi: 10.1053/j.semdp.2015.02.006. Epub 2015 Feb 24.
PEComas are mesenchymal tumors composed of histologically and immunohistochemically distinctive perivascular epithelioid cells that are characterized by the coexpression of muscle and melanogenetic markers. This group of lesions includes angiomyolipoma, clear cell "sugar" tumor of the lung and extrapulmonary sites, lymphangioleiomyomatosis, clear cell myomelanocytic tumor of the falciform ligament/ligamentum teres, and rare clear cell tumors of other anatomical sites. In the genitourinary tract, PEComas have been described in the kidney, bladder, prostate, testis, and urethra. Although most PEComas behave as benign tumors, some are potentially malignant, and criteria for malignancy have been suggested for both and renal and extrarenal lesions. Recently, the expression of cathepsin K has been demonstrated in a large number of PEComas and has been proposed as a relatively specific marker to distinguish these proliferations from the majority of human cancers. In addition, a distinctive subset of PEComas harboring TFE3 gene fusions has been reported, giving rise to a possible relationship between them and MiTF/TFE family translocation renal cell carcinomas. The genetic alterations of tuberous sclerosis complex that promote activation of the mTOR pathway have been identified in PEComas. Therapy with mTORC1 inhibitors has been shown to be effective in some cases.
PEComa是一种间质性肿瘤,由组织学和免疫组织化学上独特的血管周上皮样细胞组成,其特征是肌肉和黑色素生成标志物的共表达。这组病变包括血管平滑肌脂肪瘤、肺及肺外部位的透明细胞“糖”瘤、淋巴管平滑肌瘤病、镰状韧带/圆韧带的透明细胞肌黑色素细胞瘤,以及其他解剖部位的罕见透明细胞肿瘤。在泌尿生殖道中,PEComa已在肾脏、膀胱、前列腺、睾丸和尿道中被描述。尽管大多数PEComa表现为良性肿瘤,但有些具有潜在恶性,并且已经提出了针对肾脏和肾外病变的恶性标准。最近,组织蛋白酶K的表达已在大量PEComa中得到证实,并被提议作为一种相对特异性的标志物,以将这些增殖与大多数人类癌症区分开来。此外,已经报道了一个独特的携带TFE3基因融合的PEComa亚群,这使得它们与MiTF/TFE家族易位性肾细胞癌之间可能存在关联。在PEComa中已经鉴定出促进mTOR途径激活的结节性硬化症复合体的基因改变。在某些情况下,mTORC1抑制剂治疗已被证明是有效的。