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儿童辅酶 Q10 缺乏症:常伴有正常形态的 2C 型肌纤维。

Coenzyme Q10 deficiency in children: frequent type 2C muscle fibers with normal morphology.

机构信息

Washington University School of Medicine, Department of Neurology, 660 S. Euclid Avenue, Box 8111, St. Louis, Missouri, USA.

出版信息

Muscle Nerve. 2013 Nov;48(5):722-6. doi: 10.1002/mus.23837. Epub 2013 Sep 11.

Abstract

INTRODUCTION

Neurological disorders with low tissue coenzyme Q10 (CoQ10) levels are important to identify, as they may be treatable.

METHODS

We evaluated retrospectively clinical, laboratory, and muscle histochemistry and oxidative enzyme characteristics in 49 children with suspected mitochondrial disorders. We compared 18 with CoQ10 deficiency in muscle to 31 with normal CoQ10 values.

RESULTS

Muscle from CoQ10-deficient patients averaged 5.5-fold more frequent type 2C muscle fibers than controls (P < 0.0001). A type 2C fiber frequency of ≥ 5% had 89% sensitivity and 84% specificity for CoQ10 deficiency in this cohort. No biopsy showed active myopathy. There were no differences between groups in frequencies of mitochondrial myopathologic, clinical, or laboratory features. Multiple abnormalities in muscle oxidative enzyme activities were more frequent in CoQ10-deficient patients than in controls.

CONCLUSIONS

When a childhood mitochondrial disorder is suspected, an increased frequency of type 2C fibers in morphologically normal muscle suggests CoQ10 deficiency.

摘要

简介

低组织辅酶 Q10(CoQ10)水平的神经紊乱疾病很重要,因为它们可能是可治疗的。

方法

我们回顾性评估了 49 名疑似线粒体疾病儿童的临床、实验室和肌肉组织化学及氧化酶特征。我们比较了肌肉中 CoQ10 缺乏的 18 例患者与 CoQ10 值正常的 31 例患者。

结果

CoQ10 缺乏症患者的肌肉中,平均每例更常出现 5.5 倍的 2C 型肌纤维(P<0.0001)。在该队列中,2C 纤维频率≥5%对 CoQ10 缺乏症具有 89%的敏感性和 84%的特异性。没有活检显示活动性肌病。两组之间线粒体肌病、临床或实验室特征的频率无差异。与对照组相比,CoQ10 缺乏症患者的肌肉氧化酶活性的多种异常更为常见。

结论

当怀疑儿童患有线粒体疾病时,形态正常的肌肉中 2C 型纤维的频率增加提示 CoQ10 缺乏症。

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