Centre for Human Drug Research, Leiden, The Netherlands.
Leiden University Medical Center, Leiden, The Netherlands.
Mov Disord. 2020 Sep;35(9):1667-1674. doi: 10.1002/mds.28112. Epub 2020 Jul 2.
The most common genetic risk factor for Parkinson's disease known is a damaging variant in the GBA1 gene. The entire GBA1 gene has rarely been studied in a large cohort from a single population. The objective of this study was to assess the entire GBA1 gene in Parkinson's disease from a single large population.
The GBA1 gene was assessed in 3402 Dutch Parkinson's disease patients using next-generation sequencing. Frequencies were compared with Dutch controls (n = 655). Family history of Parkinson's disease was compared in carriers and noncarriers.
Fifteen percent of patients had a GBA1 nonsynonymous variant (including missense, frameshift, and recombinant alleles), compared with 6.4% of controls (OR, 2.6; P < 0.001). Eighteen novel variants were detected. Variants previously associated with Gaucher's disease were identified in 5.0% of patients compared with 1.5% of controls (OR, 3.4; P < 0.001). The rarely reported complex allele p.D140H + p.E326K appears to likely be a Dutch founder variant, found in 2.4% of patients and 0.9% of controls (OR, 2.7; P = 0.012). The number of first-degree relatives (excluding children) with Parkinson's disease was higher in p.D140H + p.E326K carriers (5.6%, 21 of 376) compared with p.E326K carriers (2.9%, 29 of 1014); OR, 2.0; P = 0.022, suggestive of a dose effect for different GBA1 variants.
Dutch Parkinson's disease patients display one of the largest frequencies of GBA1 variants reported so far, consisting in large part of the mild p.E326K variant and the more severe Dutch p.D140H + p.E326K founder allele. © 2020 The Authors. Movement Disorders published by Wiley Periodicals LLC. on behalf of International Parkinson and Movement Disorder Society.
已知帕金森病最常见的遗传风险因素是 GBA1 基因的有害变异。很少有研究从单一人群的大型队列中研究整个 GBA1 基因。本研究的目的是评估来自单一大型人群的帕金森病中的整个 GBA1 基因。
使用下一代测序技术评估 3402 名荷兰帕金森病患者的 GBA1 基因。将频率与荷兰对照(n = 655)进行比较。比较了携带者和非携带者的帕金森病家族史。
与对照组相比(OR,2.6;P<0.001),15%的患者存在 GBA1 非同义变异(包括错义、移码和重组等位基因),18 种新变异被检测到。在 5.0%的患者中发现了与戈谢病相关的先前报道的变体,而在对照组中为 1.5%(OR,3.4;P<0.001)。很少报道的复杂等位基因 p.D140H + p.E326K 似乎很可能是荷兰的一个创始变体,在 2.4%的患者和 0.9%的对照中发现(OR,2.7;P = 0.012)。与 p.E326K 携带者(2.9%,29/1014)相比,p.D140H + p.E326K 携带者(5.6%,21/376)的一级亲属(不包括儿童)中帕金森病的数量更高;OR,2.0;P = 0.022,提示不同 GBA1 变体存在剂量效应。
荷兰帕金森病患者表现出迄今为止报道的 GBA1 变体中最大频率之一,主要由轻度 p.E326K 变体和更严重的荷兰 p.D140H + p.E326K 创始等位基因组成。