Ramirez-Sandoval Juan C, Varela-Jimenez Ricardo, Morales-Buenrostro Luis E
Nephrology Department, National Institute of Medical Sciences and Nutrition Salvador Zubiran, Calle Vasco de Quiroga 15, Tlalpan, Sección XVI, 14000, Mexico City, Mexico.
CEN Case Rep. 2018 May;7(1):110-113. doi: 10.1007/s13730-018-0306-5. Epub 2018 Jan 17.
We report a case of capillary leak that developed during treatment of antibody-mediated rejection in a kidney transplant recipient. A 53-year-old female transplant recipient experienced an increase in serum creatinine from 1.1 to 1.8 mg/dL. Antibody-mediated rejection was diagnosed by graft biopsy. She was treated with five plasmapheresis sessions (on alternate days with albumin replacement), five doses of immunoglobulin (5 g/dose at 100 mg/kg), a single dose of rituximab (500 mg), and four doses of bortezomib on days 1, 4, 7, and 10 (1.72 mg/dose at 1.3 mg/m body surface area). During treatment, edema, slight diarrhea, pancytopenia, hypoalbuminemia, peripheral neuropathy, and postural hypotension were noted. Despite control of liquids, she presented with edema progressing to an increase of more than 10 kg body weight. Prerenal acute graft dysfunction associated with hypotension was diagnosed on day 12, heart failure or other infectious complications being discounted. On day 13, daily hemodialysis was prescribed, and a stable volume status was reached after five hemodialysis sessions. On day 20, the patient recovered diuresis and the edema and diarrhea abated, but she remained on chronic hemodialysis. After excluding other causes of distributive shock, the diagnosis of capillary leak syndrome was based on the presence of hypotension, generalized edema, and hypoalbuminemia in the absence of significant proteinuria. The concomitant presence of diarrhea, peripheral neuropathy, and pancytopenia, suggest a possible causal role for bortezomib. Awareness by clinicians of capillary leak syndrome associated with bortezomib-based treatment of AMR is paramount, despite its rarity.
我们报告了1例肾移植受者在抗体介导性排斥反应治疗期间发生毛细血管渗漏的病例。1名53岁女性肾移植受者血清肌酐从1.1mg/dL升至1.8mg/dL。经移植肾活检诊断为抗体介导性排斥反应。她接受了5次血浆置换治疗（隔日进行，同时补充白蛋白）、5剂免疫球蛋白（每剂5g，剂量为100mg/kg）、1剂利妥昔单抗（500mg）以及在第1、4、7和10天给予4剂硼替佐米（每剂1.72mg，体表面积为1.3mg/m²）。治疗期间，发现有水肿、轻度腹泻、全血细胞减少、低白蛋白血症、周围神经病变和体位性低血压。尽管控制了液体摄入，但她仍出现水肿，体重增加超过10kg。在第12天诊断为与低血压相关的肾前性急性移植肾功能障碍，排除了心力衰竭或其他感染性并发症。在第13天，开始每日进行血液透析，经过5次血液透析后达到了稳定的容量状态。在第20天，患者恢复利尿，水肿和腹泻减轻，但仍需进行长期血液透析。在排除其他分布性休克原因后，基于低血压、全身性水肿和低白蛋白血症且无明显蛋白尿，诊断为毛细血管渗漏综合征。腹泻、周围神经病变和全血细胞减少同时存在，提示硼替佐米可能起了因果作用。尽管罕见，但临床医生应认识到与基于硼替佐米治疗急性抗体介导性排斥反应相关的毛细血管渗漏综合征。
