• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

丙酸血症患者的血浆辅酶Q10水平及泛醇治疗的潜在益处

Plasma CoQ10 Status in Patients with Propionic Acidaemia and Possible Benefit of Treatment with Ubiquinol.

作者信息

Stanescu Sinziana, Belanger-Quintana Amaya, Fernández-Felix Borja Manuel, Ruiz-Sala Pedro, Alcaide Patricia, Arrieta Francisco, Martínez-Pardo Mercedes

机构信息

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.

出版信息

Antioxidants (Basel). 2022 Aug 16;11(8):1588. doi: 10.3390/antiox11081588.

DOI:10.3390/antiox11081588
PMID:36009307
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9405378/
Abstract

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.

摘要

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。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/104e/9405378/6e31f560cd34/antioxidants-11-01588-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/104e/9405378/11b80b845ab1/antioxidants-11-01588-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/104e/9405378/18a9fabe4ff9/antioxidants-11-01588-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/104e/9405378/6e31f560cd34/antioxidants-11-01588-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/104e/9405378/11b80b845ab1/antioxidants-11-01588-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/104e/9405378/18a9fabe4ff9/antioxidants-11-01588-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/104e/9405378/6e31f560cd34/antioxidants-11-01588-g003.jpg

相似文献

1
Plasma CoQ10 Status in Patients with Propionic Acidaemia and Possible Benefit of Treatment with Ubiquinol.丙酸血症患者的血浆辅酶Q10水平及泛醇治疗的潜在益处
Antioxidants (Basel). 2022 Aug 16;11(8):1588. doi: 10.3390/antiox11081588.
2
Anaplerotic therapy in propionic acidemia.丙酸血症的氨补充治疗。
Mol Genet Metab. 2017 Sep;122(1-2):51-59. doi: 10.1016/j.ymgme.2017.07.003. Epub 2017 Jul 12.
3
Ubiquinol is superior to ubiquinone to enhance Coenzyme Q10 status in older men.泛醇优于泛醌,可增强老年男性体内辅酶 Q10 的状态。
Food Funct. 2018 Nov 14;9(11):5653-5659. doi: 10.1039/c8fo00971f.
4
Comparison study of plasma coenzyme Q10 levels in healthy subjects supplemented with ubiquinol versus ubiquinone.健康受试者补充泛醇与泛醌的血浆辅酶 Q10 水平比较研究。
Clin Pharmacol Drug Dev. 2014 Jan;3(1):13-7. doi: 10.1002/cpdd.73. Epub 2013 Oct 8.
5
Reduced platelet mitochondrial respiration and oxidative phosphorylation in patients with post COVID-19 syndrome are regenerated after spa rehabilitation and targeted ubiquinol therapy.新冠后综合征患者血小板线粒体呼吸和氧化磷酸化功能降低,经温泉康复治疗和靶向泛醇治疗后得以恢复。
Front Mol Biosci. 2022 Oct 21;9:1016352. doi: 10.3389/fmolb.2022.1016352. eCollection 2022.
6
Bioavailability of Reduced Coenzyme Q10 (Ubiquinol-10) in Burn Patients.烧伤患者中还原型辅酶Q10(泛醇-10)的生物利用度。
Metabolites. 2022 Jul 1;12(7):613. doi: 10.3390/metabo12070613.
7
Successful reversal of propionic acidaemia associated cardiomyopathy: evidence for low myocardial coenzyme Q10 status and secondary mitochondrial dysfunction as an underlying pathophysiological mechanism.成功逆转丙酸血症相关心肌病:心肌辅酶 Q10 水平低和继发性线粒体功能障碍作为潜在病理生理机制的证据。
Mitochondrion. 2014 Jul;17:150-6. doi: 10.1016/j.mito.2014.07.001. Epub 2014 Jul 8.
8
Effects of Feeding Coenzyme Q10-Ubiquinol on Plasma Coenzyme Q10 Concentrations and Semen Quality in Stallions.辅酶 Q10-泛醌对种马血浆辅酶 Q10 浓度和精液质量的影响。
J Equine Vet Sci. 2021 Jan;96:103303. doi: 10.1016/j.jevs.2020.103303. Epub 2020 Oct 15.
9
The Influence of Atorvastatin, Amlodipine and Ethoxidol on Ubiquinol and Ubiquinone Endogenous Plasma Concentrations in Patients with Chronic Heart Failure.阿托伐他汀、氨氯地平和依托多醇对慢性心力衰竭患者血浆中内源性泛醇和泛醌浓度的影响。
Curr Drug Metab. 2023;24(9):635-644. doi: 10.2174/1389200224666230913133201.
10
Ubiquinol (reduced Coenzyme Q10) in patients with severe sepsis or septic shock: a randomized, double-blind, placebo-controlled, pilot trial.重度脓毒症或脓毒性休克患者使用泛醇(还原型辅酶Q10):一项随机、双盲、安慰剂对照的试点试验。
Crit Care. 2015 Jul 1;19(1):275. doi: 10.1186/s13054-015-0989-3.

引用本文的文献

1
An atypical presentation in a child with propionic acidemia? Better think twice!患有丙酸血症的儿童出现非典型表现?最好三思!
JIMD Rep. 2025 Mar 12;66(2):e12464. doi: 10.1002/jmd2.12464. eCollection 2025 Mar.
2
Prevalence of propionic acidemia in China.中国丙酸血症的流行情况。
Orphanet J Rare Dis. 2023 Sep 9;18(1):281. doi: 10.1186/s13023-023-02898-w.
3
Pathophysiological mechanisms of complications associated with propionic acidemia.丙酸血症相关并发症的病理生理机制。

本文引用的文献

1
Interorgan amino acid interchange in propionic acidemia: the missing key to understanding its physiopathology.丙酸血症中器官间氨基酸交换:理解其病理生理学的缺失关键。
Amino Acids. 2022 May;54(5):777-786. doi: 10.1007/s00726-022-03128-6. Epub 2022 Jan 30.
2
Biomarkers for drug development in propionic and methylmalonic acidemias.用于丙酸和甲基丙二酸血症药物开发的生物标志物。
J Inherit Metab Dis. 2022 Mar;45(2):132-143. doi: 10.1002/jimd.12478. Epub 2022 Jan 26.
3
Severe anemia in patients with Propionic acidemia is associated with branched-chain amino acid imbalance.
Pharmacol Ther. 2023 Sep;249:108501. doi: 10.1016/j.pharmthera.2023.108501. Epub 2023 Jul 22.
丙酸血症患者的严重贫血与支链氨基酸失衡有关。
Orphanet J Rare Dis. 2021 May 18;16(1):226. doi: 10.1186/s13023-021-01865-7.
4
Metabolic Targets of Coenzyme Q10 in Mitochondria.线粒体中辅酶Q10的代谢靶点
Antioxidants (Basel). 2021 Mar 26;10(4):520. doi: 10.3390/antiox10040520.
5
Organic acidurias: Major gaps, new challenges, and a yet unfulfilled promise.有机酸血症:主要差距、新挑战和尚未兑现的承诺。
J Inherit Metab Dis. 2021 Jan;44(1):9-21. doi: 10.1002/jimd.12254. Epub 2020 May 27.
6
Assessment of methylcitrate and methylcitrate to citrate ratio in dried blood spots as biomarkers for inborn errors of propionate metabolism.评估干血斑中的柠檬酸三甲酯和柠檬酸三甲酯与柠檬酸的比值作为丙酸代谢先天缺陷的生物标志物。
Sci Rep. 2019 Aug 26;9(1):12366. doi: 10.1038/s41598-019-48885-9.
7
Pathophysiology of propionic and methylmalonic acidemias. Part 2: Treatment strategies.丙酸血症和甲基丙二酸血症的病理生理学。第 2 部分:治疗策略。
J Inherit Metab Dis. 2019 Sep;42(5):745-761. doi: 10.1002/jimd.12128. Epub 2019 Jul 9.
8
Decreased plasma l-arginine levels in organic acidurias (MMA and PA) and decreased plasma branched-chain amino acid levels in urea cycle disorders as a potential cause of growth retardation: Options for treatment.有机酸血症(甲基丙二酸血症和丙酸血症)患者的血浆 l-精氨酸水平降低,尿素循环障碍患者的血浆支链氨基酸水平降低,可能是生长迟缓的原因:治疗选择。
Mol Genet Metab. 2019 Apr;126(4):397-405. doi: 10.1016/j.ymgme.2019.02.003. Epub 2019 Feb 25.
9
Plasma coenzyme Q status is impaired in selected genetic conditions.血浆辅酶 Q 状态在某些遗传条件下受损。
Sci Rep. 2019 Jan 28;9(1):793. doi: 10.1038/s41598-018-37542-2.
10
Bioavailability of coenzyme Q10 supplements depends on carrier lipids and solubilization.辅酶 Q10 补充剂的生物利用度取决于载体脂质和溶解。
Nutrition. 2019 Jan;57:133-140. doi: 10.1016/j.nut.2018.05.020. Epub 2018 Jun 27.