Adaptive behavior assessed by Vineland-3 as comprehensive outcome measure in vanishing white matter.

Autor: Schoenmakers DH; Department of Child Neurology, Emma's Children's Hospital, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands.; Amsterdam Leukodystrophy Center, Amsterdam Neuroscience, Cellular & Molecular Mechanisms, Amsterdam, The Netherlands.; Medicine for Society, Platform at Amsterdam UMC location University of Amsterdam, Amsterdam, The Netherlands., van Beelen I; Department of Child Neurology, Emma's Children's Hospital, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands., Voermans MMC; Department of Child Neurology, Emma's Children's Hospital, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands.; Amsterdam Leukodystrophy Center, Amsterdam Neuroscience, Cellular & Molecular Mechanisms, Amsterdam, The Netherlands., Perik D; Department of Medical Psychology, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands., Stellingwerff MD; Department of Child Neurology, Emma's Children's Hospital, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands.; Amsterdam Leukodystrophy Center, Amsterdam Neuroscience, Cellular & Molecular Mechanisms, Amsterdam, The Netherlands., Wolf NI; Department of Child Neurology, Emma's Children's Hospital, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands.; Amsterdam Leukodystrophy Center, Amsterdam Neuroscience, Cellular & Molecular Mechanisms, Amsterdam, The Netherlands., Berkhof J; Department of Epidemiology and Data Science, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands., van der Knaap MS; Department of Child Neurology, Emma's Children's Hospital, Amsterdam UMC location Vrije Universiteit, Amsterdam, The Netherlands.; Amsterdam Leukodystrophy Center, Amsterdam Neuroscience, Cellular & Molecular Mechanisms, Amsterdam, The Netherlands.; Department of Integrative Neurophysiology, Center for Neurogenomics and Cognitive Research, Vrije Universiteit, Amsterdam, The Netherlands.
Jazyk: angličtina
Zdroj: Annals of clinical and translational neurology [Ann Clin Transl Neurol] 2024 Mar; Vol. 11 (3), pp. 650-661. Date of Electronic Publication: 2024 Jan 12.
DOI: 10.1002/acn3.51985
Abstrakt: Objectives: Investigate the results and usability of the Vineland-3 as an outcome measure in vanishing white matter patients.
Methods: A 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.
Results: Adaptive 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.
Interpretation: The 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.
(© 2024 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association.)
Databáze: MEDLINE
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