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辅酶Q10缺乏症的异质性:病例研究与文献综述

Heterogeneity of coenzyme Q10 deficiency: patient study and literature review.

作者信息

Emmanuele Valentina, López Luis C, Berardo Andres, Naini Ali, Tadesse Saba, Wen Bing, D'Agostino Erin, Solomon Martha, DiMauro Salvatore, Quinzii Catarina, Hirano Michio

机构信息

Department of Neurology, Columbia University Medical Center, New York, New York, USA.

出版信息

Arch Neurol. 2012 Aug;69(8):978-83. doi: 10.1001/archneurol.2012.206.

Abstract

Coenzyme Q(10) (CoQ(10)) deficiency has been associated with 5 major clinical phenotypes: encephalomyopathy, severe infantile multisystemic disease, nephropathy, cerebellar ataxia, and isolated myopathy. Primary CoQ(10) deficiency is due to defects in CoQ(10) biosynthesis, while secondary forms are due to other causes. A review of 149 cases, including our cohort of 76 patients, confirms that CoQ(10) deficiency is a clinically and genetically heterogeneous syndrome that mainly begins in childhood and predominantly manifests as cerebellar ataxia. Coenzyme Q(10) measurement in muscle is the gold standard for diagnosis. Identification of CoQ(10) deficiency is important because the condition frequently responds to treatment. Causative mutations have been identified in a small proportion of patients.

摘要

辅酶Q(10)(CoQ(10))缺乏症与5种主要临床表型相关:脑肌病、严重婴儿多系统疾病、肾病、小脑共济失调和孤立性肌病。原发性CoQ(10)缺乏症是由于CoQ(10)生物合成缺陷所致,而继发性形式则由其他原因引起。对149例病例(包括我们的76例患者队列)的回顾证实,CoQ(10)缺乏症是一种临床和遗传异质性综合征,主要始于儿童期,主要表现为小脑共济失调。肌肉中CoQ(10)的测量是诊断的金标准。识别CoQ(10)缺乏症很重要,因为该病症通常对治疗有反应。在一小部分患者中已鉴定出致病突变。

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本文引用的文献

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Adult-onset cerebellar ataxia due to mutations in CABC1/ADCK3.成人发病小脑共济失调与 CABC1/ADCK3 基因突变相关。
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Primary and secondary CoQ(10) deficiencies in humans.原发性和继发性 CoQ10 缺乏症。
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