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伴慢性进行性眼外肌麻痹的线粒体疾病:19例临床分析

Mitochondrial disease with chronic progressive external ophthalmoplegia: clinical analysis of 19 cases.

作者信息

Kao K P

机构信息

Section of Peripheral Neurology, Veterans General Hospital-Taipei, Taiwan, R.O.C.

出版信息

Zhonghua Yi Xue Za Zhi (Taipei). 1994 Feb;53(2):95-100.

PMID:8167995
Abstract

BACKGROUND

Mitochondrial myopathy is the commonest morphological diagnosis in the patients with the syndrome of chronic progressive external ophthalmoplegia. This syndrome consists of a group of clinically heterogeneous disorders. However, systemic analysis on clinical ground is still inexplicably lacking in Chinese.

METHODS

Nineteen patients, who fulfilled the following two criteria, were collected and systematically analyzed on clinical ground. 1. Chronic progressive external ophthalmoplegia, characterized by chronic and progressive ptosis and limitation of eyeball movements, was the earliest and dominant clinical syndrome. 2. The mitochondria abnormalities were the only morphological changes on the limb muscle.

RESULTS

All patients were clinically classified into three groups. Twelve cases without extra-skeletal involvement were categorized as simple ocular myopathy; 5 cases as ophthalmoplegia plus syndrome because of the associated extra-skeletal dysfunction(s); and 2 cases as Kearns-Sayre syndrome according to the conventional triad (onset before 20 years of age, ophthalmoplegia, pigmentary degeneration of the retina) with at least one of three other major manifestations (heart block, CSF protein over 100 mg/dl, cerebellar syndrome).

CONCLUSIONS

The identification of the defects at the DNA level and determination of the phenotypic expression with basic biochemical methods as well as mitochondria DNA analysis are fundamental for more rational classification of mitochondrial myopathy. However, the classification, based on clinical criteria alone, may give the clinicians the convenience in the follow-up management of the non-skeletal disorders for an individual patient. Importantly, some of these disorders are life-threatening but treatable.

摘要

背景

线粒体肌病是慢性进行性眼外肌麻痹综合征患者最常见的形态学诊断。该综合征由一组临床异质性疾病组成。然而,在中国,基于临床的系统分析仍莫名其妙地缺乏。

方法

收集了19例符合以下两条标准的患者,并对其进行了基于临床的系统分析。1. 慢性进行性眼外肌麻痹,以慢性进行性上睑下垂和眼球运动受限为特征,是最早出现且占主导地位的临床综合征。2. 线粒体异常是肢体肌肉唯一的形态学改变。

结果

所有患者在临床上分为三组。12例无骨骼外受累的患者被归类为单纯眼肌病;5例因伴有骨骼外功能障碍而被归类为眼外肌麻痹加综合征;2例根据传统三联征(发病年龄在20岁之前、眼外肌麻痹、视网膜色素变性)以及其他三种主要表现(心脏传导阻滞、脑脊液蛋白超过100mg/dl、小脑综合征)中的至少一种被归类为卡恩斯-塞尔综合征。

结论

在DNA水平鉴定缺陷,并通过基本生化方法以及线粒体DNA分析确定表型表达,对于更合理地分类线粒体肌病至关重要。然而,仅基于临床标准的分类可能会给临床医生在对个体患者的非骨骼疾病进行随访管理时带来便利。重要的是,其中一些疾病危及生命但可治疗。

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