Oh Hye Won, Kim Tae Hyo, Cha Ra Ri, Kim Na Young, Kim Hyun Jin, Jung Woon Tae, Lee Ok Jae, Lee Jeong Hee
Korean J Gastroenterol. 2014 Nov;64(5):302-6. doi: 10.4166/kjg.2014.64.5.302.
Perivascular epithelioid cell tumors (PEComas) are unusual mesenchymal neoplasms composed of histologically and immunohistochemically distinct perivascular epithelioid cells (PECs). Although PEComas have the potential to behave in a malignant fashion, malignant PEComas arising from the retroperitoneum are extremely rare. A 68-year-old woman presented with a painful palpable mass in her left upper abdomen. Computed tomography of the abdomen showed a 9 cm sized heterogeneous mass in left para-aortic space and multiple hypervascular nodules in the liver. (18)F-fludeoxyglucose-PET/CT showed multifocal hyper-metabolic lesions in retroperitoneum, liver, and skeletal bones. Percutaneous needle biopsies were done on the retroperitoneal and hepatic mass. Both specimens were positive for human melanoma black-45 (HMB-45) on histological and immunohistochemical staining which was compatible with PEComas. Herein, we report a rare case of retroperitoneal PEComa with multiple metastases involving liver and bone at initial diagnosis that exhibited aggressive behavior and resulted in a devastating prognosis.
血管周上皮样细胞肿瘤(PEComas)是一种不常见的间叶性肿瘤,由组织学和免疫组织化学上独特的血管周上皮样细胞(PEC)组成。虽然PEComas有恶变的可能,但起源于腹膜后的恶性PEComas极为罕见。一名68岁女性因左上腹可触及的疼痛性肿块就诊。腹部计算机断层扫描显示左主动脉旁间隙有一个9厘米大小的不均匀肿块,肝脏有多个高血供结节。(18)F-氟脱氧葡萄糖-PET/CT显示腹膜后、肝脏和骨骼有多灶性高代谢病变。对腹膜后和肝脏肿块进行了经皮穿刺活检。组织学和免疫组织化学染色显示,两个标本的人黑素瘤黑色45(HMB-45)均为阳性,这与PEComas相符。在此,我们报告一例罕见的腹膜后PEComa,初诊时即有肝和骨的多发转移,表现出侵袭性生物学行为,预后极差。