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

Daphne H. Schoenmakers; Irene van Beelen; Marije M.C. Voermans; Denise Perik; Menno D. Stellingwerff; Nicole I. Wolf; Johannes Berkhof; Marjo S. van der Knaap

doi:10.1002/acn3.51985

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

Daphne H. Schoenmakers, Irene van Beelen, Marije M.C. Voermans, Denise Perik, Menno D. Stellingwerff, Nicole I. Wolf, Johannes Berkhof, Marjo S. van der Knaap^*

^*Corresponding author for this work

Research output: Contribution to Journal › Article › Academic › peer-review

Abstract

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.

Original language	English
Pages (from-to)	650-661
Number of pages	12
Journal	Annals of Clinical and Translational Neurology
Volume	11
Issue number	3
Early online date	12 Jan 2024
DOIs	https://doi.org/10.1002/acn3.51985
Publication status	Published - Mar 2024

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© 2024 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association.

Funding

The following authors of this publication are members of the European Reference Network for Rare Neurological Diseases (ERN‐RND) – Project ID No 739510: N.I. Wolf, M.S. van der Knaap. Part of this study was supported by a grant of ERN‐RND.

Funders	Funder number
ERN-RND	739510

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title = "Adaptive behavior assessed by Vineland-3 as comprehensive outcome measure in vanishing white matter",

abstract = "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.",

author = "Schoenmakers, \{Daphne H.\} and \{van Beelen\}, Irene and Voermans, \{Marije M.C.\} and Denise Perik and Stellingwerff, \{Menno D.\} and Wolf, \{Nicole I.\} and Johannes Berkhof and \{van der Knaap\}, \{Marjo S.\}",

note = "Publisher Copyright: {\textcopyright} 2024 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association.",

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doi = "10.1002/acn3.51985",

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AU - van Beelen, Irene

AU - Voermans, Marije M.C.

AU - Perik, Denise

AU - Stellingwerff, Menno D.

AU - Wolf, Nicole I.

AU - Berkhof, Johannes

AU - van der Knaap, Marjo S.

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

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