Abstract
In the current study, we examined adaptive skills and trajectories over time in 257 individuals with Angelman syndrome (AS) using the Vineland Adaptive Behavior Scales, 2nd Edition. Multilevel linear models were used to examine differences between molecular subtypes over time, from one year to 13 years of age, in the adaptive domains of communication, daily living skills, socialization and motor skills. Individuals with non-deletion subtypes typically demonstrated a higher level of adaptive skills compared to those with deletion subtypes. Statistically significant growth was observed in all adaptive domains through at least early adolescence. Individuals with AS should continue to receive developmental services and educational supports through adolescence and into adulthood given the slow rates of growth being observed across adaptive domains.
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Data Availability
The data sets used and/or analyzed during this study are available from the corresponding author on reasonable request.
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Acknowledgements
We extend our deepest appreciation to all of the individuals with AS and their family members who participated in the AS Natural History Study. We are also grateful to the site principal investigators and the study teams at each site for coordinating recruitment and collecting data.
Funding
This study was supported by NIH U54 RR019478 (awarded to Arthur L. Beaudet) from the National Center for Research Resources (NCRR) and NIH U54 HD061222 (awarded to Alan Percy) from the National Institute of Child Health and Human Development (NICHD), both components of the National Institutes of Health. A special thanks to Foundation for Angelman Syndrome Therapeutics (FAST) for providing funding and the Angelman Biomarkers and Outcome Measures Alliance (A-BOM) for their support of the preparation of this manuscript.
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All authors contributed to the study conception and design. WHT and LB received funding for the study. Data were collected by SP, RBW, LH, LN, RH and AS. Material preparation and data analyses were performed by AG. The first draft of the manuscript was prepared by SNP and AG and reviewed by AS and AW. All authors reviewed subsequent drafts and read and approved the final manuscript.
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WHT and AW have received research support and/or compensation for consulting from Ovid Therapeutics, F. Hoffman-LaRoche, and Ionis. WHT and AS have also received support/compensation from Biogen.
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This study was approved by the Institutional Review Board at each study site.
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Gwaltney, A., Potter, S.N., Peters, S.U. et al. Adaptive Skills of Individuals with Angelman Syndrome Assessed Using the Vineland Adaptive Behavior Scales, 2nd Edition. J Autism Dev Disord (2023). https://doi.org/10.1007/s10803-023-06090-8
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DOI: https://doi.org/10.1007/s10803-023-06090-8