Adaptive behavior assessed by Vineland-3 as comprehensive outcome measure in vanishing white matter
ANNALS OF CLINICAL AND TRANSLATIONAL NEUROLOGY(2024)
Abstract
ObjectivesInvestigate the results and usability of the Vineland-3 as an outcome measure in vanishing white matter patients.MethodsA cross-sectional investigation of the Vineland-3 based on interviews with caregivers, the Health Utilities Index, and the modified Rankin Scale in 64 vanishing white matter patients.ResultsAdaptive behavior measured with the Vineland-3 is impaired in the vast majority of vanishing white matter patients and significantly impacts daily life. Typically, the daily living skills domain is most severely affected and the socialization domain is the least affected. Based on the metric properties and the clinical relevance, the standard scores for the daily living skills domain and Adaptive Behavior Composite have the best properties to be used as an outcome measure.InterpretationThe Vineland-3 appears to be a useful outcome measure to explore and quantify complex cognitive, behavioral, and psychiatric impairments affecting daily functioning in vanishing white matter. Further research should address the longitudinal evaluation of this tool and its additional value to standard neuropsychological and clinical examination.
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