Adaptive Skills of Individuals with Angelman Syndrome Assessed Using the Vineland Adaptive Behavior Scales, 2nd Edition.
Angela Gwaltney, Sarah Nelson Potter, Sarika U Peters and 8 others
PMID 37581718WHAT IT FOUND
Children with Angelman syndrome showed growth in adaptive skills through age 13, but expressive communication remained the slowest area.
Deletion subtypes scored lower than non-deletion subtypes. Early intervention should focus on alternative communication methods.
Key findings
01Individuals with Angelman syndrome demonstrated statistically significant growth in adaptive skills across all Vineland-II subdomains throughout childhood and into early adolescence, with growth rates slowing as age increased.
02Expressive communication was the subdomain with the slowest overall growth rate for all molecular subtypes, and individuals with deletion subtypes had significantly lower expressive communication scores at age 6 compared to non-deletion subtypes.
03At age 6, adaptive skills varied widely by molecular subtype, with deletion groups showing age-equivalent scores around 12 to 18 months, while non-deletion groups showed scores between 18 and 38 months.
STILL TO COME
How it was doneWhat they foundWhat it means for PTsWhat it means for OTsWhat it means for SLPs
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What it does not show
The study used the Vineland-II, which does not account for augmentative and alternative communication (AAC), potentially underestimating expressive communication skills. Floor effects were significant in domestic and community subdomains, limiting the accuracy of age-equivalent scores in these areas. ImpD and UPD groups were combined due to small sample sizes, preventing separate analysis of these subtypes. Data were collected between 2006 and 2014, and the Vineland-3, which includes AAC credit, is now the standard. Variability in the number of visits per participant (1 to 9) may affect the precision of trajectory predictions.
Declared interests
The study was supported by the NIH Rare Diseases Clinical Research Network. No specific financial conflicts of interest with commercial entities are reported in the provided text.
The easy way to misread this
Do not interpret low expressive communication scores as a lack of potential. The Vineland-II does not credit AAC or non-verbal communication, so these scores may underestimate actual functional communication abilities. Always assess alternative modes of communication directly.