van Tilburg Sander J, Koçer Selin, Geissler Judy, van Rijs Wouter, Tio-Gillen Anne P, Tanck Michael W T, Fokkink Willem-Jan R, van Doorn Pieter A, Jacobs Bart C, Nagelkerke Sietse Q, Huizinga Ruth
Department of Immunology, Erasmus MC, University Medical Center Rotterdam, Dr. Molewaterplein 40, 3015 GD, Rotterdam, The Netherlands.
Department of Blood Cell Research, Sanquin Research Institute, University of Amsterdam, Amsterdam, The Netherlands.
J Neurol. 2025 Jul 1;272(7):487. doi: 10.1007/s00415-025-13216-8.
Fc-gamma receptors (FcγRs) are important for the effector functions of immunoglobulin G (IgG) and are therefore expected to play a role in the pathophysiology of Guillain-Barré syndrome (GBS). The FCGR2/3 locus, which encodes low-to-medium-affinity FcγRs, contains extensive genetic variation. We hypothesized that genetic variation in the FCGR2/3 locus influences GBS susceptibility, muscle weakness, outcomes, and the pharmacokinetics of intravenous immunoglobulin (IVIg).
Copy number variation and single nucleotide polymorphisms in the FCGR2/3 locus were studied using multiplex ligation-dependent probe amplification (MLPA). The study cohort consisted of 467 GBS patients and 919 healthy controls of European descent. Severe weakness was defined as an MRC sum score < 40 at nadir. The increase in serum IgG one or two weeks after start of IVIg treatment was determined.
No significant associations were found between genetic variation in the FCGR2/3 locus and susceptibility to GBS. However, in patients with an antecedent Campylobacter jejuni infection, a higher frequency of three or more FCGR3A copies was observed compared to healthy controls (p = 0.023). FCGR3A copy numbers were also associated with more severe disease (OR = 2.02; 95% CI = 1.00-4.12), even after correcting for age and positive C. jejuni serology. No association was found between FCGR2/3 variants and the ability to walk unaided in time-to-event analyses. In addition, the pharmacokinetics of IVIg were not affected by genetic variation in the FCGR2/3 locus.
Overall, FCGR2/3 polymorphisms are not associated with susceptibility to GBS or response to IVIg treatment. However, associations may exist in specific subgroups, as demonstrated in patients with a preceding C. jejuni infection who more frequently carry a duplication in FCGR3A.
Fc-γ受体(FcγRs)对于免疫球蛋白G(IgG)的效应功能很重要,因此预计其在吉兰-巴雷综合征(GBS)的病理生理学中发挥作用。编码低至中等亲和力FcγRs的FCGR2/3基因座存在广泛的基因变异。我们假设FCGR2/3基因座的基因变异会影响GBS易感性、肌肉无力、预后以及静脉注射免疫球蛋白(IVIg)的药代动力学。
使用多重连接依赖探针扩增(MLPA)研究FCGR2/3基因座中的拷贝数变异和单核苷酸多态性。研究队列包括467例GBS患者和919例欧洲血统的健康对照。严重肌无力定义为最低点时医学研究委员会(MRC)总分<40。测定IVIg治疗开始后一或两周血清IgG的增加情况。
未发现FCGR2/3基因座的基因变异与GBS易感性之间存在显著关联。然而,在先前有空肠弯曲菌感染的患者中,与健康对照相比,观察到三个或更多FCGR3A拷贝的频率更高(p = 0.023)。即使校正年龄和空肠弯曲菌血清学阳性后,FCGR3A拷贝数也与更严重的疾病相关(OR = 2.02;95%CI = 1.00 - 4.12)。在事件发生时间分析中,未发现FCGR2/3变异与独立行走能力之间存在关联。此外,FCGR2/3基因座的基因变异不影响IVIg的药代动力学。
总体而言,FCGR2/3多态性与GBS易感性或IVIg治疗反应无关。然而,在特定亚组中可能存在关联,如先前有空肠弯曲菌感染的患者中FCGR3A重复的频率更高所证明的那样。