Suppr超能文献

肌阵挛性癫痫伴破碎红纤维的无症状患者的视网膜神经元丧失。

Retinal Neuronal Loss in Visually Asymptomatic Patients With Myoclonic Epilepsy With Ragged-Red Fibers.

机构信息

Department of Visual Neurosciences (RPN, DM), Singapore Eye Research Institute, The Academia, Singapore; Ophthalmology and Visual Sciences Program (RPN, DM), Duke-NUS Medical School, Singapore; Departments of Ophthalmology, Biochemistry and Genetics (PR, AC, VP, PA-B and DM), Angers University Hospital, Angers, France; Department of Neuro-Ophthalmology (DM), Singapore National Eye Centre, Singapore; and Department of Surgery (Ophthalmology) (HM), Melbourne Medical School, University of Melbourne, Melbourne, Australia.

出版信息

J Neuroophthalmol. 2019 Mar;39(1):18-22. doi: 10.1097/WNO.0000000000000690.

Abstract

BACKGROUND

Myoclonic epilepsy with ragged-red fibers (MERRF, OMIM, #545000) is a rare neurological condition mostly caused by the m.8344A>G mitochondrial DNA pathogenic variant, which can variably affect multiple tissues, including the retina and optic nerve. We report detection of visually asymptomatic neuroretinal loss in 3 patients with genetically confirmed MERRF, using spectral domain optical coherence tomography (SD-OCT).

METHODS

All patients underwent a complete ophthalmic examination including assessments of visual acuity, color vision, pupillary reactions, extraocular movements, applanation tonometry, slit-lamp, and dilated fundus examinations. Standard automated perimetry or Goldmann kinetic perimetry was performed, as well as fundus photographs and SD-OCT of the optic nerve head and macula.

RESULTS

Despite the absence of visual symptoms in all patients, and normal visual acuity and visual fields in 1 patient, the 3 genetically confirmed patients (point mutations m.8344A>G; age range: 18-62 years) with MERRF-related neurological manifestations, displayed thinning of the retinal nerve fiber layer and variable alterations of the macular ganglion cell complex.

CONCLUSIONS

Visually asymptomatic patients with genetically confirmed MERRF can display features of structural neuroretinal loss, quantifiable with SD-OCT. Further investigations are needed to establish whether OCT can assess early neurodegeneration in MERRF.

摘要

背景

肌阵挛性癫痫伴破碎红纤维(MERRF,OMIM,#545000)是一种罕见的神经疾病,主要由 m.8344A>G 线粒体 DNA 致病性变异引起,该变异可不同程度地影响包括视网膜和视神经在内的多种组织。我们报告了使用谱域光学相干断层扫描(SD-OCT)在 3 名经基因证实的 MERRF 患者中检测到无症状性神经视网膜丧失。

方法

所有患者均接受全面眼科检查,包括视力、色觉、瞳孔反应、眼球运动、眼压测量、裂隙灯和眼底检查。进行标准自动视野检查或 Goldmann 动态视野检查,以及眼底照相和视神经头和黄斑的 SD-OCT。

结果

尽管所有患者均无视觉症状,且 1 名患者视力正常,但这 3 名具有 MERRF 相关神经表现的基因确诊患者(点突变 m.8344A>G;年龄范围:18-62 岁),显示视网膜神经纤维层变薄和黄斑神经节细胞复合体的不同改变。

结论

经基因证实的无症状 MERRF 患者可出现结构神经视网膜丧失的特征,可通过 SD-OCT 定量。需要进一步研究以确定 OCT 是否可以评估 MERRF 中的早期神经退行性变。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验