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GAIN 登记研究——多器官自身免疫和自身炎症患者的一项新前瞻性研究。

The GAIN Registry - a New Prospective Study for Patients with Multi-organ Autoimmunity and Autoinflammation.

机构信息

Division Methods in Clinical Epidemiology, Institute of Medical Biometry and Statistics, Faculty of Medicine and Medical Center, University of Freiburg, Freiburg, Germany.

Institute for Immunodeficiency, Center for Chronic Immunodeficiency (CCI), Faculty of Medicine, Medical Center, University of Freiburg, Breisacher Str. 115, 79106, Freiburg, Germany.

出版信息

J Clin Immunol. 2023 Aug;43(6):1289-1301. doi: 10.1007/s10875-023-01472-0. Epub 2023 Apr 21.

Abstract

Patient registries are a very important and essential tool for investigating rare diseases, as most physicians only see a limited number of cases during their career. Diseases of multi-organ autoimmunity and autoinflammation are especially challenging, as they are characterized by diverse clinical phenotypes and highly variable expressivity. The GAIN consortium (German multi-organ Auto Immunity Network) developed a dataset addressing these challenges. ICD-11, HPO, and ATC codes were incorporated to document various clinical manifestations and medications with a defined terminology. The GAIN dataset comprises detailed information on genetics, phenotypes, medication, and laboratory values. Between November 2019 and July 2022, twelve centers from Europe have registered 419 patients with multi-organ autoimmunity or autoinflammation. The median age at onset of symptoms was 13 years (IQR 3-28) and the median delay from onset to diagnosis was 5 years (IQR 1-14). Of 354 (84.5%) patients who were genetically tested, 248 (59.2%) had a defined monogenetic cause. For 87 (20.8%) patients, no mutation was found and for 19 (4.5%), the result was pending. The most common gene affected was NFkB1 (48, 11.5%), and the second common was CTLA4 (40, 9.5%), both genetic patient groups being fostered by specific research projects within GAIN. The GAIN registry may serve as a valuable resource for research in the inborn error of immunity community by providing a platform for etiological and diagnostic research projects, as well as observational trials on treatment options.

摘要

患者登记处是研究罕见疾病的非常重要和必要的工具,因为大多数医生在其职业生涯中只看到有限数量的病例。多器官自身免疫和自身炎症性疾病尤其具有挑战性,因为它们具有不同的临床表现和高度可变的表达。GAIN 联盟(德国多器官自身免疫网络)开发了一个数据集来应对这些挑战。ICD-11、HPO 和 ATC 代码被纳入其中,以使用定义的术语记录各种临床表现和药物。GAIN 数据集包含有关遗传学、表型、药物和实验室值的详细信息。在 2019 年 11 月至 2022 年 7 月期间,来自欧洲的 12 个中心登记了 419 名患有多器官自身免疫或自身炎症性疾病的患者。症状发作的中位年龄为 13 岁(IQR 3-28),从发作到诊断的中位延迟为 5 年(IQR 1-14)。在接受基因检测的 354 名(84.5%)患者中,有 248 名(59.2%)有明确的单基因病因。87 名(20.8%)患者未发现突变,19 名(4.5%)患者结果待定。受影响最常见的基因是 NFkB1(48 名,11.5%),其次是 CTLA4(40 名,9.5%),这两个基因患者群体都是 GAIN 内部的特定研究项目促成的。GAIN 登记处可以作为先天免疫错误社区研究的有价值资源,为病因学和诊断研究项目以及治疗选择的观察性试验提供平台。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1087/10354141/ae00daa13351/10875_2023_1472_Fig1_HTML.jpg

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