Epithelioid Angiosarcoma Arising in the Non-function Arteriovenous Fistula in a Renal Transplant Recipient-Case Report and Literature Review

發生於腎移植病患之無功能動靜脈廔管處之惡性類上皮血管肉瘤－病例報告及文獻回顧

蔡岳錤(Yueh-Chi Tsai) ； 陳伊呈(I-Chen Chen) ； 賴志昇(Chih-Sheng Lai) ； 呂俊德(Chen-Te Lu) ； 顏榮信(Jung-Hsing Yen) ； 宋定宇(Ding-Yu Song)

《臺灣整形外科醫學會雜誌》 24卷1期 (2015/03) Pp. 51-58

摘要

背景：血管肉瘤是一種罕見但極具侵襲性惡性腫瘤且預後不佳，發生率約佔所有肉瘤之百分之一。文獻報告指出在免疫抑制的移植病患身上，血管肉瘤可從許多不同的部位長出。目的及目標：特別的是，目前為止文獻報告中僅有九個血管肉瘤之病例是發生在腎臟移植術後病患的無功能性動靜脈廔管；本文章將呈現第十位個案，亦是第一位華人病例報告，並於文中討論其可能之成因。材料及方法：文章中描述一位五十七歲男性接受腎臟移植後罹患由無功能性動靜脈廔管長出之類上皮血管肉瘤，該病患之藥物史和腫瘤之臨床表徵、影像學發現、手術術式、病理特性及預後都將於文章中予以討論。另外，本文中將針對此不常見疾病進行相關文獻之回顧。結果：包含本個案，目前只有十位由無功能性動靜脈廔管長出之類上皮血管肉瘤病例報告；於疾病診斷時僅有兩位被發現腫瘤已轉移。文中之病患接受腫瘤廣泛性切除並以左大腿前外側游離皮瓣予以重建。結論：腎臟移植後病患罹患由無功能性動靜脈廔管長出之類上皮血管肉瘤是罕見的但卻有生命威脅性的。免疫抑制劑及廔管擾流可能是造成血管肉瘤產生之成因；由於移植病人之日益增加，此個案提醒外科醫師需對於非頭皮血管肉瘤之增加提高警覺。

並列摘要

Background: Angiosarcoma is a rare but aggressive malignant tumor with a poor prognosis, accounting for only 1% of sarcoma. In the immunosuppressed transplant recipients, angiosarcoma has been reported at various sites. Aim and Objectives: Specifically, to date, there were only 9 cases of angiosarcoma arising in the non-function arteriovenous (AV) fistula after renal transplant. In this article, the 10_(th) case was presented and possible histogenesis was discussed. Material and Methods: Here, we described the case of a 57-year-old male renal transplant recipient who suffered from an epithelioid angiosarcoma developing in a non-function AV fistula. The distinguishing clinical finding, image studies, medical history, surgical procedures, pathology feature and follow-up outcomes were reported. Review of the literature with regards to this uncommon entity is presented. Results: Only 10 cases of angiosarcoma arising in the AV fistula have been reported, including this case. Metastasis was noted at diagnosis only in 2 cases. Wide excision and reconstruction with an anteriolateral thigh (ALT) free flap was performed in this case. Conclusion: Epithelioid angiosarcoma developing in an AV fistula in the immunosuppressed post-transplant patient is a rare but life-threatening event. Immunosuppression and aberrant flow through a fistula contribute to the genesis of angiosarcoma. This case alerts surgeons to the increase in non-scalp angiosarcoma, as the number of renal transplant patients has increased recently.

並列關鍵字

epithelioid angiosarcoma ； arteriovenous fistula ； immunosuppression