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Mov Disord. 2018 Dec;33(12):1857-1870. doi: 10.1002/mds.27527. Epub 2018 Oct 24.
2
Genotype-Phenotype Relations for the Parkinson's Disease Genes Parkin, PINK1, DJ1: MDSGene Systematic Review.帕金森病基因 Parkin、PINK1、DJ1 的基因型-表型关系:MDSGene 系统评价。
Mov Disord. 2018 May;33(5):730-741. doi: 10.1002/mds.27352. Epub 2018 Apr 11.
3
LRRK2 kinase in Parkinson's disease.帕金森病中的LRRK2激酶
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Genetic Forms of Parkinson's Disease.帕金森病的遗传形式
Semin Neurol. 2017 Apr;37(2):135-146. doi: 10.1055/s-0037-1601567. Epub 2017 May 16.

在一项基于英国大型人群的帕金森病研究中对孟德尔突变进行的遗传分析。

Genetic analysis of Mendelian mutations in a large UK population-based Parkinson's disease study.

机构信息

Department of Clinical and Movement Neurosciences, UCL Queen Square Institute of Neurology, London, UK.

UCL Movement Disorders Centre, University College London, London, UK.

出版信息

Brain. 2019 Sep 1;142(9):2828-2844. doi: 10.1093/brain/awz191.

DOI:10.1093/brain/awz191
PMID:31324919
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6735928/
Abstract

Our objective was to define the prevalence and clinical features of genetic Parkinson's disease in a large UK population-based cohort, the largest multicentre prospective clinico-genetic incident study in the world. We collected demographic data, Movement Disorder Society Unified Parkinson's Disease Rating Scale scores, and Montreal Cognitive Assessment scores. We analysed mutations in PRKN (parkin), PINK1, LRRK2 and SNCA in relation to age at symptom onset, family history and clinical features. Of the 2262 participants recruited to the Tracking Parkinson's study, 424 had young-onset Parkinson's disease (age at onset ≤ 50) and 1799 had late onset Parkinson's disease. A range of methods were used to genotype 2005 patients: 302 young-onset patients were fully genotyped with multiplex ligation-dependent probe amplification and either Sanger and/or exome sequencing; and 1701 late-onset patients were genotyped with the LRRK2 'Kompetitive' allele-specific polymerase chain reaction assay and/or exome sequencing (two patients had missing age at onset). We identified 29 (1.4%) patients carrying pathogenic mutations. Eighteen patients carried the G2019S or R1441C mutations in LRRK2, and one patient carried a heterozygous duplication in SNCA. In PRKN, we identified patients carrying deletions of exons 1, 4 and 5, and P113Xfs, R275W, G430D and R33X. In PINK1, two patients carried deletions in exon 1 and 5, and the W90Xfs point mutation. Eighteen per cent of patients with age at onset ≤30 and 7.4% of patients from large dominant families carried pathogenic Mendelian gene mutations. Of all young-onset patients, 10 (3.3%) carried biallelic mutations in PRKN or PINK1. Across the whole cohort, 18 patients (0.9%) carried pathogenic LRRK2 mutations and one (0.05%) carried an SNCA duplication. There is a significant burden of LRRK2 G2019S in patients with both apparently sporadic and familial disease. In young-onset patients, dominant and recessive mutations were equally common. There were no differences in clinical features between LRRK2 carriers and non-carriers. However, we did find that PRKN and PINK1 mutation carriers have distinctive clinical features compared to young-onset non-carriers, with more postural symptoms at diagnosis and less cognitive impairment, after adjusting for age and disease duration. This supports the idea that there is a distinct clinical profile of PRKN and PINK1-related Parkinson's disease. We estimate that there are approaching 1000 patients with a known genetic aetiology in the UK Parkinson's disease population. A small but significant number of patients carry causal variants in LRRK2, SNCA, PRKN and PINK1 that could potentially be targeted by new therapies, such as LRRK2 inhibitors.

摘要

我们的目标是在一个大型英国人群队列中定义遗传帕金森病的流行率和临床特征,这是世界上最大的多中心前瞻性临床遗传发病研究。我们收集了人口统计学数据、运动障碍协会统一帕金森病评定量表评分和蒙特利尔认知评估评分。我们分析了 PRKN(parkin)、PINK1、LRRK2 和 SNCA 中的突变与发病年龄、家族史和临床特征的关系。在追踪帕金森氏症的 2262 名参与者中,424 名患有早发性帕金森病(发病年龄≤50 岁),1799 名患有晚发性帕金森病。我们使用多种方法对 2005 名患者进行基因分型:302 名早发性患者通过多重连接依赖性探针扩增进行了全面基因分型,并用 Sanger 和/或外显子测序;1701 名晚发性患者通过 LRRK2“竞争性”等位基因特异性聚合酶链反应检测和/或外显子测序进行基因分型(两名患者的发病年龄缺失)。我们发现 29 名(1.4%)患者携带致病性突变。18 名患者携带 LRRK2 的 G2019S 或 R1441C 突变,1 名患者携带 SNCA 的杂合性重复。在 PRKN 中,我们发现患者携带外显子 1、4 和 5 的缺失以及 P113Xfs、R275W、G430D 和 R33X。在 PINK1 中,两名患者携带外显子 1 和 5 的缺失以及 W90Xfs 点突变。发病年龄≤30 岁的患者中有 18%和来自大型显性家族的患者中有 7.4%携带致病性孟德尔基因突变。所有早发性患者中,有 10 名(3.3%)患者携带 PRKN 或 PINK1 的双等位基因突变。在整个队列中,有 18 名(0.9%)患者携带致病性 LRRK2 突变,1 名(0.05%)患者携带 SNCA 重复。在明显散发性和家族性疾病患者中,LRRK2 G2019S 的负担很大。在早发性患者中,显性和隐性突变同样常见。LRRK2 携带者与非携带者之间的临床特征没有差异。然而,我们确实发现 PRKN 和 PINK1 突变携带者与早发性非携带者相比具有独特的临床特征,在调整年龄和疾病持续时间后,诊断时更易出现姿势症状,认知障碍更少。这支持了 PRKN 和 PINK1 相关帕金森病存在独特临床特征的观点。我们估计在英国帕金森病人群中,有近 1000 名患者具有已知的遗传病因。一小部分但具有统计学意义的患者携带 LRRK2、SNCA、PRKN 和 PINK1 中的因果变异,这些变异可能成为新疗法的靶点,如 LRRK2 抑制剂。