Fukushima N, Saito S, Sakata Y, Sawa Y
Department of Therapeutics for End-Stage Organ Dysfunction, Osaka University Graduate School of Medicine, Osaka, Japan.
Transplant Proc. 2013 Oct;45(8):3144-6. doi: 10.1016/j.transproceed.2013.08.082.
We herein report a case of putative everolimus-associated chylothorax in a cardiac transplant recipient. A 17-year-old Japanese boy with dilated cardiomyopathy and severe cardiac failure requiring left ventricular assist support was determined to be a cardiac transplant candidate in 1992. He underwent overseas heart transplantation in Houston, Texas in October 1992. He was subsequently treated with immunosuppression therapy: Cyclosporine (CSA), azathioprine, and prednisolone (PRD). After several acute rejection episodes requiring steroid therapy, intravascular ultrasonography revealed a moderate degree of transplant coronary arterial vasculopathy (TCAV) with 50% stenosis in 2003. He underwent coronary stenting twice; the immunosuppressive regimen was converted to CSA, mycophenolate mofetil, everolimus (EVL), and PRD in 2006. TCAV has not progressed since then. In October 2008, chest x-ray showed bilateral pleural effusion. As we thought that the pleural effusion was caused by cardiac dysfunction due to moderate mitral regurgitation and TCAV as well as renal impairment, he was treated with diuretics and digoxin. However, the pleural effusion progressed gradually associated with exertional dyspnea and moderate edema of his lower legs. Chest computed tomography showed massive bilateral pleural effusions without evidence of malignancy in 2011. A pleural tap in 2011 revealed chylothorax. Although mammalian target of rapamycin inhibitors were major drugs for lymphoangioleimyomatosis, we believed that the chylothorax was associated with EVL. EVL was discontinued in March 2011: the chylothorax spontaneously resolved in November 2011.
我们在此报告一例心脏移植受者中疑似与依维莫司相关的乳糜胸病例。一名17岁患有扩张型心肌病和严重心力衰竭且需要左心室辅助支持的日本男孩于1992年被确定为心脏移植候选者。1992年10月,他在得克萨斯州休斯顿接受了海外心脏移植手术。随后他接受了免疫抑制治疗:环孢素(CSA)、硫唑嘌呤和泼尼松龙(PRD)。在经历了几次需要类固醇治疗的急性排斥反应后,血管内超声检查显示2003年存在中度移植冠状动脉血管病变(TCAV),狭窄率为50%。他接受了两次冠状动脉支架置入术;免疫抑制方案于2006年转换为CSA、霉酚酸酯、依维莫司(EVL)和PRD。从那时起,TCAV未再进展。2008年10月,胸部X线显示双侧胸腔积液。由于我们认为胸腔积液是由中度二尖瓣反流、TCAV以及肾功能损害导致的心脏功能障碍引起的,因此他接受了利尿剂和地高辛治疗。然而,胸腔积液逐渐进展,并伴有劳力性呼吸困难和小腿中度水肿。2011年胸部计算机断层扫描显示双侧大量胸腔积液,未发现恶性肿瘤迹象。2011年的胸腔穿刺显示为乳糜胸。尽管雷帕霉素靶蛋白抑制剂是淋巴管平滑肌瘤病的主要药物,但我们认为乳糜胸与依维莫司有关。依维莫司于2011年3月停用:乳糜胸于2011年11月自行消退。