Department of Paediatrics and Child Health, Steve Biko Academic Hospital, University of Pretoria, PO Box 32781, Totiusdal, Pretoria 0134, South Africa.
J Inherit Metab Dis. 2010 Dec;33 Suppl 3:S95-104. doi: 10.1007/s10545-009-9031-8. Epub 2010 Feb 5.
Mitochondrial disorders are frequently encountered inherited diseases characterized by unexplained multisystem involvement with a chronic, intermittent, or progressive nature. The objective of this paper is to describe the profile of patients with mitochondrial disorders in South Africa. Patients with possible mitochondrial disorders were accessed over 10 years. Analyses for respiratory chain and pyruvate dehydrogenase complex enzymes were performed on muscle. A diagnosis of a mitochondrial disorder was accepted only if an enzyme activity was deficient. Sixty-three patients were diagnosed with a mitochondrial disorder, including 40 African, 20 Caucasian, one mixed ancestry, and two Indian patients. The most important findings were the difference between African patients and other ethnicities: respiratory chain enzyme complexes CI+III or CII+III deficiencies were found in 52.5% of African patients, being of statistical significance (p value = 0.0061). They also presented predominantly with myopathy (p value = 0.0018); the male:female ratio was 1:1.2. Twenty-five (62.5%) African patients presented with varying degrees of a myopathy accompanied by a myopathic face, high palate, and scoliosis. Fourteen of these 25 also had ptosis and/or progressive external ophthalmoplegia. No patients of other ethnicities presented with this specific myopathic phenotype. Caucasian patients (16/20) presented predominantly with central nervous system involvement. Of the South African pediatric neurology patients, Africans are more likely to present with myopathy and CII+III deficiency, and Caucasian patients are more likely to present with encephalopathy or encephalomyopathy.
线粒体疾病是一类常见的遗传性疾病,其特征为多系统受累,呈慢性、间歇性或进行性。本文旨在描述南非线粒体疾病患者的特征。在 10 多年的时间里,对疑似线粒体疾病的患者进行了评估。对肌肉中的呼吸链和丙酮酸脱氢酶复合物酶进行了分析。只有当酶活性缺乏时,才会被诊断为线粒体疾病。共诊断出 63 例线粒体疾病患者,包括 40 例非洲人、20 例白种人、1 例混血儿和 2 例印度人。最重要的发现是非洲患者与其他种族之间的差异:52.5%的非洲患者存在呼吸链酶复合物 CI+III 或 CII+III 缺乏,这具有统计学意义(p 值=0.0061)。他们也主要表现为肌病(p 值=0.0018);男女比例为 1:1.2。25 名(62.5%)非洲患者表现出不同程度的肌病,伴有肌病面容、高腭和脊柱侧凸。其中 14 例还伴有上睑下垂和/或进行性眼外肌麻痹。没有其他种族的患者出现这种特定的肌病表型。白种人患者(20 例中的 16 例)主要表现为中枢神经系统受累。在南非儿科神经病学患者中,非洲人更可能表现为肌病和 CII+III 缺乏,而白种人更可能表现为脑病或脑肌病。