Stanescu Sinziana, Belanger-Quintana Amaya, Fernández-Felix Borja Manuel, Ruiz-Sala Pedro, Alcaide Patricia, Arrieta Francisco, Martínez-Pardo Mercedes
Unidad de Enfermedades Metabólicas, Hospital Universitario Ramón y Cajal, IRYCIS, Crta de Colmenar Viejo, km 9, 100, PC 28034 Madrid, Spain.
Unidad de Bioestadística Clínica, Instituto Ramon y Cajal de Investigación Sanitaria, Hospital Universitario Ramón y Cajal, Crta de Colmenar Viejo, km 9, 100, PC 28034 Madrid, Spain.
Antioxidants (Basel). 2022 Aug 16;11(8):1588. doi: 10.3390/antiox11081588.
Propionic acidaemia (PA) is an innate error of metabolism involving a deficiency in the enzyme propionyl-CoA carboxylase. Better control of acute decompensation episodes together with better treatment and monitoring have improved the prognosis of patients with this problem. However, long-term complications can arise in those in whom good metabolic control is achieved, the result of mitochondrial dysfunction caused by deficient anaplerosis, increased oxidative stress, and reduced antioxidative capacity. Coenzyme Q10 (CoQ10) is a nutritional supplement that has a notable antioxidative effect and has been shown to improve mitochondrial function. The present prospective, interventional study examines the plasma concentration of CoQ10 in patients with PA, their tolerance of such supplementation with ubiquinol, and its benefits. Seven patients with PA (aged 2.5 to 20 years, 4 males) received supplements of CoQ10 in the form of ubiquinol (10 mg/kg/day for 6 months). A total of 6/7 patients showed reduced plasma CoQ10 concentrations that normalized after supplementation with ubiquinol (p-value < 0.001), which was well tolerated. Urinary citrate levels markedly increased during the study (p-value: 0.001), together with elevation of citrate/methlycitrate ratio (p-value: 0.03). No other significant changes were seen in plasma or urine biomarkers of PA. PA patients showed a deficiency of plasma CoQ10, which supplementation with ubiquinol corrected. The urinary excretion of Krebs cycle intermediate citrate and the citrate/methylcitrate ratio significantly increased compared to the baseline, suggesting improvement in anaplerosis. This treatment was well tolerated and should be further investigated as a means of preventing the chronic complications associated with likely multifactorial mitochondrial dysfunction in PA.
丙酸血症(PA)是一种先天性代谢缺陷,涉及丙酰辅酶A羧化酶缺乏。更好地控制急性失代偿发作以及更好的治疗和监测改善了患有该疾病患者的预后。然而,在代谢控制良好的患者中可能会出现长期并发症,这是由回补反应不足、氧化应激增加和抗氧化能力降低导致的线粒体功能障碍所致。辅酶Q10(CoQ10)是一种具有显著抗氧化作用的营养补充剂,已被证明可改善线粒体功能。本项前瞻性干预研究考察了PA患者的血浆CoQ10浓度、他们对泛醇补充剂的耐受性及其益处。7例PA患者(年龄2.5至20岁,4例男性)接受了泛醇形式的CoQ10补充剂(10mg/kg/天,持续6个月)。7例患者中有6例血浆CoQ10浓度降低,在补充泛醇后恢复正常(p值<0.001),且耐受性良好。研究期间尿柠檬酸水平显著升高(p值:0.001),同时柠檬酸/甲基柠檬酸比值升高(p值:0.03)。PA的血浆或尿液生物标志物未见其他显著变化。PA患者表现出血浆CoQ10缺乏,补充泛醇可纠正这一情况。与基线相比,三羧酸循环中间产物柠檬酸的尿排泄量及柠檬酸/甲基柠檬酸比值显著增加,提示回补反应有所改善。这种治疗耐受性良好,应作为预防PA中可能与多因素线粒体功能障碍相关的慢性并发症的一种手段进行进一步研究。