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Developing Meaningful Score Differences for the Bayley-4 and Vineland-3 in Angelman Syndrome Using a Delphi Panel.

Sonya Powers1, Katherine N Anderson2, Wen-Hann Tan2

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Summary

Caregivers established meaningful score differences (MSDs) for the Bayley Scales of Infant Development, Fourth Edition (Bayley-4) and Vineland Adaptive Behavior Scales, Third Edition (Vineland-3) in individuals with Angelman syndrome (AS). These MSDs reflect changes impacting daily life and quality of life for AS patients and their families.

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Angelman syndromeclinical outcome assessmentmeaningful change

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Area of Science:

  • Developmental Psychology
  • Clinical Assessment
  • Rare Diseases

Background:

  • Angelman syndrome (AS) is a rare genetic disorder affecting development.
  • Standardized assessments like the Bayley Scales of Infant Development, Fourth Edition (Bayley-4) and Vineland Adaptive Behavior Scales, Third Edition (Vineland-3) are used to track development in individuals with AS.
  • Establishing meaningful score differences (MSDs) is crucial for interpreting assessment results in the context of daily functioning.

Purpose of the Study:

  • To develop within-patient meaningful score differences (MSDs) for the Bayley-4 and Vineland-3 assessments.
  • To incorporate the caregiver perspective in defining what constitutes a meaningful change for individuals with Angelman syndrome (AS).

Main Methods:

  • A Delphi method was employed with a panel of 19 caregivers of individuals with AS.
  • Caregivers rated vignettes describing varying baseline functioning levels to determine MSDs for Bayley-4 and Vineland-3 subscales.
  • An iterative process with three rating rounds and two discussion rounds was used to achieve consensus.

Main Results:

  • High agreement rates (≥70%) were achieved for final MSD ratings across Bayley-4 and Vineland-3 subscales.
  • MSD thresholds were not fixed cut-offs but varied based on the individual's baseline functioning level.
  • The study identified preliminary MSD estimates for interpreting changes in individuals with AS.

Conclusions:

  • The Delphi Panel method effectively integrated caregiver insights to establish preliminary MSDs for the Bayley-4 and Vineland-3 in individuals with AS.
  • These caregiver-defined MSDs provide a valuable framework for understanding meaningful within-person change in AS, considering diverse baseline functioning levels.