ArticleValue in health : the journal of the International Society for Pharmacoeconomics and Outcomes Research2026
Caregiver Meaningful Score Differences and Meaningful Score Regions for the Observer-Reported Communication Ability Measure for Individuals with Angelman Syndrome.
Article in Value in health : the journal of the International Society for Pharmacoeconomics and Outcomes Research, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.
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1 citing paper in PubMed.
- Evaluating different scoring algorithms to assess communication ability for individuals living with Angelman syndrome: the Observer-Reported Communication Ability (ORCA) measure.Journal of patient-reported outcomes · 2026Article
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11 authors.
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Abstract
objectivesThis study presents a novel method for estimating meaningful score differences (MSDs) and meaningful score regions (MSRs) for the Observer-Reported Communication Ability (ORCA) Measure for individuals with Angelman syndrome (AS).
methodsThe AS Natural History Study collected longitudinal data on an individual's communication abilities on the ORCA Measure, Vineland Adaptive Behavior Scales-3, and the Bayley Scales of Infant and Toddler Development-4. In a separate Delphi-based study, caregivers provided MSDs and MSRs for the Vineland-3 and Bayley-4 scores. Leveraging the strong correlations between ORCA and these measures, mixed models with random intercepts were used to estimate ORCA MSDs and MSRs based on the previously derived Vineland-3/Bayley-4 MSDs and MSRs.
resultsThe ORCA Measure was completed by 192 caregivers, with 113 also completing the Bayley-4 and 159 completing the Vineland-3. ORCA T-score MSRs for low, intermediate, and high communication ability were estimated for expressive, receptive, and pragmatic communication domains using the Vineland-3 and Bayley-4 previously derived MSDs and MSRs. ORCA MSDs varied depending on the MSR and ranged from 0.38-4.55 (based on Vineland-3) and 1.79-4.62 (based on Bayley-4).
conclusionsWe present a novel anchor-based methodological approach for deriving MSD and MSR thresholds for clinical outcome assessments. Although these MSD and MSR estimates for the ORCA measure are preliminary, our methods offer a strategy for establishing meaningful change thresholds in rare disease populations in which traditional longitudinal methods may be challenging to implement.
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