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线粒体脑肌病伴乳酸血症和卒中样发作(MELAS):线粒体tRNA亮氨酸1(MTTL1)A3243G MELAS突变的多代影响

MELAS: A Multigenerational Impact of the MTTL1 A3243G MELAS Mutation.

作者信息

Prasad M, Narayan B, Prasad A N, Rupar C A, Levin S, Kronick J, Ramsay D, Tay K Y, Prasad C

出版信息

Can J Neurol Sci. 2014 Mar;41(2):210-9. doi: 10.1017/s0317167100016607.

Abstract

BACKGROUND

the maternally inherited MTTL1 A3243G mutation in the mitochondrial genome causes MelaS (Mitochondrial encephalopathy lactic acidosis with Stroke-like episodes), a condition that is multisystemic but affects primarily the nervous system. Significant intra-familial variation in phenotype and severity of disease is well recognized.

METHODS

retrospective and ongoing study of an extended family carrying the MTTL1 A3243G mutation with multiple symptomatic individuals. tissue heteroplasmy is reviewed based on the clinical presentations, imaging studies, laboratory findings in affected individuals and pathological material obtained at autopsy in two of the family members.

RESULTS

there were seven affected individuals out of thirteen members in this three generation family who each carried the MTTL1 A3243G mutation. the clinical presentations were varied with symptoms ranging from hearing loss, migraines, dementia, seizures, diabetes, visual manifestations, and stroke like episodes. three of the family members are deceased from MelaS or to complications related to MelaS.

CONCLUSIONS

the results of the clinical, pathological and radiological findings in this family provide strong support to the current concepts of maternal inheritance, tissue heteroplasmy and molecular pathogenesis in MelaS. neurologists (both adult and paediatric) are the most likely to encounter patients with MelaS in their practice. genetic counselling is complex in view of maternal inheritance and heteroplasmy. newer therapeutic options such as arginine are being used for acute and preventative management of stroke like episodes.

摘要

背景

线粒体基因组中母系遗传的MTTL1 A3243G突变会导致线粒体脑肌病伴乳酸血症和卒中样发作(MelaS),这是一种多系统疾病,但主要影响神经系统。疾病表型和严重程度在家族内部存在显著差异,这一点已得到充分认识。

方法

对一个携带MTTL1 A3243G突变的大家庭中多名有症状个体进行回顾性和持续性研究。根据临床表现、影像学检查、受影响个体的实验室检查结果以及两名家庭成员尸检时获得的病理材料,对组织异质性进行评估。

结果

在这个三代家庭的13名成员中,有7人受影响,他们均携带MTTL1 A3243G突变。临床表现多样,症状包括听力丧失、偏头痛、痴呆、癫痫、糖尿病、视觉表现和卒中样发作。其中三名家庭成员死于MelaS或与MelaS相关的并发症。

结论

该家庭临床、病理和影像学检查结果有力支持了目前关于MelaS的母系遗传、组织异质性和分子发病机制的概念。神经科医生(成人和儿科)在临床实践中最有可能遇到MelaS患者。鉴于母系遗传和异质性,遗传咨询较为复杂。精氨酸等新的治疗选择正被用于卒中样发作的急性和预防性治疗。

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