Alessandra Girardi, Linda Abetz-Webb, Katja Rudell, Paldeep S Atwal, Alexander Fay
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Psychometric properties of the Infantile Neuroaxonal Dystrophy Rating Scale.
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.
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.
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