Value in health : the journal of the International Society for Pharmacoeconomics and Outcomes Research · 2026
Developing Meaningful Score Differences for the Bayley-4 and Vineland-3 in Angelman Syndrome Using a Delphi Panel.
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Powers Sonya, Anderson Katherine N, Tan Wen-Hann, Tillmann Julian, Daniel Mark, Thurm Audrey, Farmer Cristan, Clinch Susanne, Wheeler Anne C, Sadhwani Anjali, Angelman Syndrome Meaningful Change Delphi Working Group
Аннотация
OBJECTIVES: This study aimed to develop within-patient meaningful score differences (MSDs) on the Bayley Scales of Infant Development, Fourth Edition (Bayley-4), and the Vineland Adaptive Behavior Scales, Third Edition (Vineland-3), for individuals with Angelman syndrome (AS).
METHODS: A Delphi method, involving a panel of 19 caregivers of individuals with AS, was used to establish MSDs for Bayley-4 and Vineland-3 growth scale values. MSD was defined as the smallest change that would noticeably affect the daily functioning of an individual with AS or family quality of life in a way that was important to the individual with AS or their family. For each subscale of the Bayley-4 and Vineland-3, the panel was presented with 2 to 4 vignettes describing varying levels of baseline functioning and asked to select an MSD from a range of potential values. An iterative process involving 3 rounds of ratings and 2 rounds of discussion was used to build consensus. The median caregiver rating from round 3 was retained as the final recommended MSD value for each vignette.
RESULTS: Final MSD ratings for the 5 subscales of Bayley-4 and 10 subscales of the Vineland-3 had an agreement rate of 70% or higher. MSD thresholds for each subscale were not single cutoffs, but rather reflected a range of MSD values dependent on the level of baseline functioning.
CONCLUSIONS: The Delphi panel method incorporates the caregiver perspective to provide preliminary estimates of what constitutes meaningful within-person change on the Bayley-4 and Vineland-3 in individuals with AS with various levels of baseline functioning.
METHODS: A Delphi method, involving a panel of 19 caregivers of individuals with AS, was used to establish MSDs for Bayley-4 and Vineland-3 growth scale values. MSD was defined as the smallest change that would noticeably affect the daily functioning of an individual with AS or family quality of life in a way that was important to the individual with AS or their family. For each subscale of the Bayley-4 and Vineland-3, the panel was presented with 2 to 4 vignettes describing varying levels of baseline functioning and asked to select an MSD from a range of potential values. An iterative process involving 3 rounds of ratings and 2 rounds of discussion was used to build consensus. The median caregiver rating from round 3 was retained as the final recommended MSD value for each vignette.
RESULTS: Final MSD ratings for the 5 subscales of Bayley-4 and 10 subscales of the Vineland-3 had an agreement rate of 70% or higher. MSD thresholds for each subscale were not single cutoffs, but rather reflected a range of MSD values dependent on the level of baseline functioning.
CONCLUSIONS: The Delphi panel method incorporates the caregiver perspective to provide preliminary estimates of what constitutes meaningful within-person change on the Bayley-4 and Vineland-3 in individuals with AS with various levels of baseline functioning.