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迟发性发病的Leber遗传性视神经病变:20例患者的临床和分子特征

Leber's hereditary optic neuropathy with late disease onset: clinical and molecular characteristics of 20 patients.

作者信息

Dimitriadis Konstantin, Leonhardt Miriam, Yu-Wai-Man Patrick, Kirkman Matthew Anthony, Korsten Alex, De Coo Irenaeus F, Chinnery Patrick Francis, Klopstock Thomas

出版信息

Orphanet J Rare Dis. 2014 Oct 23;9:158. doi: 10.1186/s13023-014-0158-9.

Abstract

BACKGROUND

Leber's hereditary optic neuropathy (LHON) is a mitochondrial disease that typically causes bilateral blindness in young men. Here we describe the clinical and molecular characteristics of 20 patients with disease onset after the age of 50 years (late onset-LHON).

METHODS

From a cohort of 251 affected and 277 unaffected LHON carriers, we identified 20 patients with onset of visual loss after the age of 50 years. Using structured questionnaires, data including basic demographic details, age of onset, progression of visual loss and severity as well as exposure to possible environmental triggers including alcohol, smoking and illicit drugs were retrospectively collected. Groups were compared using the Mann-Whitney-U-Test for two independent groups of sampled data.

RESULTS

The proportion of late onset-LHON in our cohort was 8% (20 patients, 15 males, 5 females). The mtDNA mutations m.11778G > A and m.3460G > A were found in 16 and 4 patients, respectively. Among 89 asymptomatic carriers above the age of 50 years (28 males, 61 females), the mtDNA mutations m.11778G > A, m.3460G > A and m.14484 T > C were found in 60, 12 and 17 carriers, respectively. Late onset-LHON patients had significantly higher mean cumulative tobacco and alcohol consumption compared with unaffected carriers. However, there was no significant difference between late onset- and typical LHON patients with regard to daily tobacco and weekly alcohol consumption before disease onset.

CONCLUSION

As already shown for typical LHON, alcohol consumption and smoking are important trigger factors also for the late manifestation. LHON should be considered in the differential diagnosis of subacute blindness even in older patients.

摘要

背景

Leber遗传性视神经病变(LHON)是一种线粒体疾病,通常导致年轻男性双眼失明。在此,我们描述了20例50岁以后发病的患者(迟发性LHON)的临床和分子特征。

方法

在251例受影响的和277例未受影响的LHON携带者队列中,我们确定了20例50岁以后出现视力丧失的患者。使用结构化问卷,回顾性收集包括基本人口统计学细节、发病年龄、视力丧失进展和严重程度以及接触可能的环境触发因素(包括酒精、吸烟和非法药物)等数据。使用Mann-Whitney-U检验对两组独立抽样数据进行组间比较。

结果

我们队列中迟发性LHON的比例为8%(20例患者,15例男性,5例女性)。分别在16例和4例患者中发现线粒体DNA突变m.11778G>A和m.3460G>A。在89例50岁以上的无症状携带者(28例男性,61例女性)中,分别在60例、12例和17例携带者中发现线粒体DNA突变m.11778G>A、m.3460G>A和m.14484T>C。与未受影响的携带者相比,迟发性LHON患者的平均累计烟草和酒精消费量显著更高。然而,在发病前,迟发性LHON患者与典型LHON患者在每日烟草消费量和每周酒精消费量方面没有显著差异。

结论

正如典型LHON已显示的那样,饮酒和吸烟也是迟发性LHON的重要触发因素。即使在老年患者中,亚急性失明的鉴别诊断也应考虑LHON。

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