Psychometric properties of the Infantile Neuroaxonal Dystrophy Rating Scale.

IF 4.3 2区医学 Q1 CLINICAL NEUROLOGY

Developmental Medicine and Child Neurology Pub Date : 2025-09-19 DOI:10.1111/dmcn.16506

Alessandra Girardi, Linda Abetz-Webb, Katja Rudell, Paldeep S Atwal, Alexander Fay

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Abstract

Aim: To assess the psychometric characteristics of the Infantile Neuroaxonal Dystrophy Rating Scale (INAD-RS).

Method: We retrospectively analysed interim data from the INAD Natural History Study using clinicians' ratings (n = 39) of patients with infantile neuroaxonal dystrophy (INAD). Data were analysed to explore the internal consistency, test-retest reliability, known group validity, and longitudinal changes of the INAD-RS.

Results: The analysis identified good internal consistency, convergent validity, and test-retest reliability across scale domains assessing gross motor, fine motor, bulbar, ocular, and temporo-frontal functions, whereas the domain of autonomic nervous system function contributed weakly to the overall INAD-RS score. Furthermore, preliminary evidence suggested that INAD-RS total scores discriminated among clinical phenotypes and was sensitive to change over time.

Interpretation: The psychometric properties of the INAD-RS showed construct validity and reliability for five out of six domains (autonomic nervous system excluded). The scale is a promising instrument for evaluating children with INAD in clinical research and clinical practice. Although promising, further evidence is needed to refine the scale and support its suitability for assessing disease progression in clinical trials and clinical practice.

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婴儿神经轴突营养不良评定量表的心理测量特征。

目的：评价婴儿神经轴索营养不良评定量表（INAD-RS）的心理测量特征。方法：我们使用临床医生对婴儿神经轴突营养不良（INAD）患者的评分（n = 39），回顾性分析INAD自然史研究的中期数据。对数据进行分析，探讨INAD-RS的内部一致性、重测信度、已知组效度和纵向变化。结果：分析发现，在评估大运动、精细运动、球、眼和颞额功能的量表域中，有良好的内部一致性、收敛效度和重测信度，而自主神经系统功能域对整体INAD-RS评分的贡献较弱。此外，初步证据表明，INAD-RS总分在临床表型中存在差异，且随时间变化敏感。解释：INAD-RS的心理测量特性在6个领域（自主神经系统除外）中的5个领域显示了构念效度和信度。该量表在临床研究和临床实践中具有较好的应用前景。虽然有希望，但需要进一步的证据来完善该量表，并支持其在临床试验和临床实践中评估疾病进展的适用性。

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

Developmental Medicine and Child Neurology 医学-临床神经学

CiteScore

7.80

自引率

13.20%

发文量

338

审稿时长

3-6 weeks

期刊介绍： Wiley-Blackwell is pleased to publish Developmental Medicine & Child Neurology (DMCN), a Mac Keith Press publication and official journal of the American Academy for Cerebral Palsy and Developmental Medicine (AACPDM) and the British Paediatric Neurology Association (BPNA). For over 50 years, DMCN has defined the field of paediatric neurology and neurodisability and is one of the world’s leading journals in the whole field of paediatrics. DMCN disseminates a range of information worldwide to improve the lives of disabled children and their families. The high quality of published articles is maintained by expert review, including independent statistical assessment, before acceptance.