Vernon Hilary J, Sperati C John, King Joshua D, Poretti Andrea, Miller Neil R, Sloan Jennifer L, Cameron Andrew M, Myers Donna, Venditti Charles P, Valle David
McKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University, 733 N. Broadway, BRB 529, Baltimore, MD, 21205, USA,
J Inherit Metab Dis. 2014 Nov;37(6):899-907. doi: 10.1007/s10545-014-9730-7. Epub 2014 Jun 25.
End stage kidney disease is a well-known complication of methylmalonic acidemia (MMA), and can be treated by dialysis, kidney transplant, or combined kidney-liver transplant. While liver and/or kidney transplantation in MMA may reduce the risk of metabolic crisis and end-organ disease, it does not fully prevent disease-related complications. We performed detailed metabolite and kinetic analyses in a 28-year-old patient with mut (0) MMA who underwent hemodialysis for 6 months prior to receiving a combined liver/kidney transplant. A single hemodialysis session led to a 54 % reduction in plasma methylmalonic acid and yielded a plasma clearance of 103 ml/min and VD0.48 L/kg, which approximates the total body free water space. This was followed by rapid reaccumulation of methylmalonic acid over 24 h to the predialysis concentration in the plasma. Following combined liver/kidney transplantation, the plasma methylmalonic acid was reduced to 3 % of pre-dialysis levels (6,965 ± 1,638 (SD) μmol/L and 234 ± 100 (SD) μmol/L) but remained >850× higher than the upper limit of normal (0.27 ± 0.08 (SD) μmol/L). Despite substantial post-operative metabolic improvement, the patient developed significant neurologic complications including acute worsening of vision in the setting of pre-existing bilateral optic neuropathy, generalized seizures, and a transient, focal leukoencephalopathy. Plasma methylmalonic acid was stable throughout the post-operative course. The biochemical parameters exhibited by this patient further define the whole body metabolism of methylmalonic acid in the setting of dialysis and subsequent combined liver/kidney transplant.
终末期肾病是甲基丙二酸血症(MMA)的一种众所周知的并发症,可通过透析、肾移植或肝肾联合移植进行治疗。虽然MMA患者进行肝和/或肾移植可能会降低代谢危机和终末器官疾病的风险,但并不能完全预防与疾病相关的并发症。我们对一名28岁的mut(0)MMA患者进行了详细的代谢物和动力学分析,该患者在接受肝肾联合移植前接受了6个月的血液透析。单次血液透析可使血浆甲基丙二酸降低54%,血浆清除率为103 ml/min,分布容积为0.48 L/kg,接近全身自由水空间。随后,甲基丙二酸在24小时内迅速重新蓄积至透析前血浆浓度。肝肾联合移植后,血浆甲基丙二酸降至透析前水平的3%(6,965±1,638(标准差)μmol/L和234±100(标准差)μmol/L),但仍比正常上限(0.27±0.08(标准差)μmol/L)高850倍以上。尽管术后代谢有显著改善,但患者仍出现了严重的神经并发症,包括在已有双侧视神经病变的情况下视力急性恶化、全身性癫痫发作和短暂性局灶性白质脑病。术后整个过程中血浆甲基丙二酸水平稳定。该患者表现出的生化参数进一步明确了透析及随后肝肾联合移植情况下甲基丙二酸的全身代谢情况。