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多发性硬化症中与莱伯遗传性视神经病变相关的线粒体DNA突变

Leber's hereditary optic neuropathy mitochondrial DNA mutations in multiple sclerosis.

作者信息

Kellar-Wood H, Robertson N, Govan G G, Compston D A, Harding A E

机构信息

University of Cambridge Neurology Unit, Addenbrooke's Hospital, United Kingdom.

出版信息

Ann Neurol. 1994 Jul;36(1):109-12. doi: 10.1002/ana.410360121.

Abstract

The observation of a multiple sclerosis (MS)-like illness in patients, particularly women, who carry the most common Leber's hereditary optic neuropathy mitochondrial DNA (mtDNA) mutation may indicate a contributory role for mitochondrial genes in genetic susceptibility to MS. We screened 307 unrelated MS patients, ascertained from population surveys, for the pathogenic Leber's hereditary optic neuropathy mutations at positions 11778 and 3460 of mtDNA, and also studied 20 patients with prominent and early optic nerve involvement. In addition, these 307 patients and 129 control subjects were investigated for the base change at position 13708, which has been suggested to play a role in the pathogenesis of Leber's hereditary optic neuropathy. Neither of the pathogenic mtDNA mutations occurred in the unselected MS patients. The 13708 base change was present in MS patients at a frequency similar to that in healthy control subjects. Three of the patients selected on the basis of severe optic nerve involvement had either the 11778 (one) or 3460 (two) mutations associated with Leber's hereditary optic neuropathy. All were women and none had affected relatives. We conclude that these mutations do not contribute to genetically determined susceptibility in typical MS patients, although a mitochondrial genetic component in the etiology of MS remains possible. A subgroup of MS patients, particularly females with severe bilateral visual failure due to optic neuropathy, may harbor a Leber's hereditary optic neuropathy mutation and we suggest that mtDNA analysis is appropriate in these patients.

摘要

在携带最常见的Leber遗传性视神经病变线粒体DNA(mtDNA)突变的患者,尤其是女性患者中观察到类似多发性硬化症(MS)的疾病,这可能表明线粒体基因在MS的遗传易感性中起作用。我们对通过人群调查确定的307名无亲缘关系的MS患者进行了筛查,以检测mtDNA第11778和3460位点的致病性Leber遗传性视神经病变突变,并且还研究了20名有明显早期视神经受累的患者。此外,对这307名患者和129名对照受试者进行了第13708位点碱基变化的研究,该变化被认为在Leber遗传性视神经病变的发病机制中起作用。在未选择的MS患者中均未出现致病性mtDNA突变。MS患者中第13708位点碱基变化的频率与健康对照受试者相似。根据严重视神经受累情况选择的3名患者中,有1名携带与Leber遗传性视神经病变相关的11778突变,2名携带3460突变。所有患者均为女性,且均无患病亲属。我们得出结论,这些突变对典型MS患者的遗传易感性没有影响,尽管MS病因中线粒体遗传成分仍有可能存在。MS患者的一个亚组,尤其是因视神经病变导致严重双侧视力丧失的女性,可能携带Leber遗传性视神经病变突变,我们建议对这些患者进行mtDNA分析。

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