TY - JOUR
T1 - Adaptive behavior assessed by Vineland-3 as comprehensive outcome measure in vanishing white matter
AU - Schoenmakers, Daphne H.
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.
N1 - Funding Information: 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. Publisher Copyright: © 2024 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association.
PY - 2024/3
Y1 - 2024/3
N2 - 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.
AB - 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.
UR - http://www.scopus.com/inward/record.url?scp=85182222568&partnerID=8YFLogxK
U2 - https://doi.org/10.1002/acn3.51985
DO - https://doi.org/10.1002/acn3.51985
M3 - Article
C2 - 38217081
SN - 2328-9503
VL - 11
SP - 650
EP - 661
JO - Annals of clinical and translational neurology
JF - Annals of clinical and translational neurology
IS - 3
ER -