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神经病、共济失调和色素性视网膜炎综合征

Neuropathy, Ataxia, and Retinitis Pigmentosa Syndrome.

作者信息

Finsterer Josef

机构信息

Neurology & Neurophysiology Center, Vienna, Austria.

出版信息

J Clin Neuromuscul Dis. 2023 Mar 1;24(3):140-146. doi: 10.1097/CND.0000000000000422.

Abstract

OBJECTIVES

To provide an overview about the phenotype, genotype, treatment, and outcome of neuropathy, ataxia, and retinitis pigmentosa (NARP) syndrome.

METHODS

Systematic review by application of appropriate search terms.

RESULTS

NARP syndrome is a syndromic mitochondrial disorder due to pathogenic variants in MT-ATP6. The canonical phenotypic features of NARP syndrome include proximal muscle weakness, axonal neuropathy, cerebellar ataxia, and retinitis pigmentosa. Noncanonical phenotypic features in NARP include epilepsy, cerebral or cerebellar atrophy, optic atrophy, cognitive impairment, dementia, sleep apnea syndrome, hearing impairment, renal insufficiency, and diabetes. So far, 10 pathogenic variants in MT-ATP6 have been associated with NARP, NARP-like syndrome, or NARP/maternally inherited Leigh overlap syndrome. Most pathogenic MT-ATP6 variants are missense, but a few truncating pathogenic variants have been reported. The most common variant responsible for NARP is the transversion m.8993T>G. Only symptomatic treatment for NARP syndrome is available. In most of the cases, patients die prematurely. Patients with late-onset NARP survive longer.

CONCLUSIONS

NARP is a rare, syndromic, monogenic mitochondrial disorder due to pathogenic variants in MT-ATP6. The nervous system and the eyes are most commonly affected. Although only symptomatic treatment is available, the outcome is usually fair.

摘要

目的

概述神经病变、共济失调和色素性视网膜炎(NARP)综合征的表型、基因型、治疗及预后情况。

方法

通过应用适当的检索词进行系统综述。

结果

NARP综合征是一种由MT - ATP6基因致病性变异导致的综合征性线粒体疾病。NARP综合征的典型表型特征包括近端肌无力、轴索性神经病变、小脑共济失调和色素性视网膜炎。NARP的非典型表型特征包括癫痫、大脑或小脑萎缩、视神经萎缩、认知障碍、痴呆、睡眠呼吸暂停综合征、听力障碍、肾功能不全和糖尿病。到目前为止,MT - ATP6基因中的10种致病性变异已与NARP、NARP样综合征或NARP/母系遗传的 Leigh重叠综合征相关。大多数致病性MT - ATP6变异为错义变异,但也有少数截短致病性变异的报道。导致NARP的最常见变异是颠换m.8993T>G。NARP综合征仅能进行对症治疗。在大多数病例中,患者过早死亡。迟发型NARP患者存活时间更长。

结论

NARP是一种由MT - ATP6基因致病性变异引起的罕见的、综合征性、单基因线粒体疾病。神经系统和眼睛最常受累。尽管仅能进行对症治疗,但预后通常尚可。

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