Rasch-Built Overall Disability Scale for IgM-Associated Polyneuropathy With and Without Anti-MAG Antibodies: IgM-RODS

IF 3.2 3区医学 Q1 CLINICAL NEUROLOGY

Journal of the Peripheral Nervous System Pub Date : 2025-09-23 DOI:10.1111/jns.70059

Tatiana Hamadeh, Johannes P. M. van de Mortel, Janneke G. J. Hoeijmakers, David R. Cornblath, Alexander F. J. E. Vrancken, Catharina G. Faber, Nicolette C. Notermans, Perry T. C. van Doormaal, Ingemar S. J. Merkies, the IMAGiNe Consortium

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引用次数: 0

Abstract

Background and Aim

IgM monoclonal gammopathy-associated polyneuropathy with(out) anti-myelin associated glycoprotein (±anti-MAG) is a rare immune-mediated disease that may cause severe limitations in daily activities and quality of life. The absence of a systematic comparison between patients with/without anti-MAG IgM polyneuropathy, no disease-specific functional metric, and lack of international consensus regarding assessment and treatment of these patients are factors obstructing future clinical trials. Therefore, it was decided to develop an interval Rasch-built activity/participation scale specifically for IgM polyneuropathy ±anti-MAG (IgM-RODS) and examine its clinimetric properties.

Methods

A pre-phase IgM-RODS questionnaire containing 146 activity/participation items, based on the WHO International Classification of Functioning, Disability and Health, was completed by participants (≥ 18 years) of the IMAGiNe observational registry that fulfilled international criteria for IgM-polyneuropathy ±anti-MAG. Data was subjected to Rasch analyses, and reliability/validity studies were performed as well.

Results

The pre-RODS data of 259 subjects (originating from 8 different countries) underwent quality assessment, and 244 remaining records were submitted to the Rasch model, evidencing the model's expectations. Based on requirements like exceeding fit residuals, misfit statistics, item bias, local dependency, and less face validity, we systematically removed items until the final 36-item IgM-RODS fulfilled all Rasch requirements and showed acceptable test–retest reliability, cross-cultural, construct and discriminant validity, and unidimensionality. Compared to the Inflammatory-RODS, the IgM-RODS showed lower standard errors across the metric, indicating greater sensitivity.

Interpretation

The 36-item IgM-RODS is a disease-specific interval measure suitable for detecting functional deficits in patients with IgM-polyneuropathy ±anti-MAG. Future studies are needed to determine its responsiveness.

Abstract Image

查看原文本刊更多论文

rasch建立的igm相关多发性神经病变的总体残疾量表，有或没有抗mag抗体：IgM-RODS。

背景和目的：IgM单克隆γ -病变相关多神经病变伴（out）抗髓鞘相关糖蛋白（±anti-MAG）是一种罕见的免疫介导疾病，可导致日常活动和生活质量的严重限制。缺乏对患有/不患有抗mag IgM多神经病变患者的系统比较，没有疾病特异性功能指标，以及缺乏关于这些患者的评估和治疗的国际共识，这些都是阻碍未来临床试验的因素。因此，我们决定开发一种专门针对IgM多发性神经病变±抗mag （IgM- rods）的间歇rasch构建的活动/参与量表，并检查其临床特性。方法：IgM-RODS前期问卷包含146个活动/参与项目，基于WHO国际功能、残疾和健康分类，由IMAGiNe观察登记的参与者（≥18岁）完成，符合igm -多发性神经病变±抗mag的国际标准。数据进行了Rasch分析，并进行了信度/效度研究。结果：对来自8个不同国家的259名受试者的pre-RODS数据进行了质量评估，并将剩余的244份记录提交给Rasch模型，证明了模型的预期。基于超拟合残差、不拟合统计、项目偏差、局部依赖和较少的面效度等要求，我们系统地删除了项目，直到最终的36个IgM-RODS满足所有Rasch要求，并显示出可接受的重测信度、跨文化信度、结构效度和判别效度以及单维性。与Inflammatory-RODS相比，IgM-RODS在整个度量中显示出更低的标准误差，表明更高的灵敏度。解释：36项IgM-RODS是一种疾病特异性间隔测量，适用于检测igm -多发性神经病变±抗mag患者的功能缺陷。需要进一步的研究来确定其响应性。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Journal of the Peripheral Nervous System 医学-临床神经学

CiteScore

6.10

自引率

7.90%

发文量

审稿时长

>12 weeks

期刊介绍： The Journal of the Peripheral Nervous System is the official journal of the Peripheral Nerve Society. Founded in 1996, it is the scientific journal of choice for clinicians, clinical scientists and basic neuroscientists interested in all aspects of biology and clinical research of peripheral nervous system disorders. The Journal of the Peripheral Nervous System is a peer-reviewed journal that publishes high quality articles on cell and molecular biology, genomics, neuropathic pain, clinical research, trials, and unique case reports on inherited and acquired peripheral neuropathies. Original articles are organized according to the topic in one of four specific areas: Mechanisms of Disease, Genetics, Clinical Research, and Clinical Trials. The journal also publishes regular review papers on hot topics and Special Issues on basic, clinical, or assembled research in the field of peripheral nervous system disorders. Authors interested in contributing a review-type article or a Special Issue should contact the Editorial Office to discuss the scope of the proposed article with the Editor-in-Chief.