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Parent Description of Anxiety in Angelman Syndrome

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Abstract

Anxiety is being increasingly identified in Angelman syndrome (AS). Qualitative questions and quantitative assessments were used to evaluate for anxiety in 50 subjects with AS. In-person evaluations assessed behaviors concerning for anxiety and circumstances wherein they occurred. Caregivers completed anxiety and other behavioral rating scales. Caregiver responses were categorized and compared to items from anxiety rating scales. The most common behavioral manifestation of anxiety was “aggression.” The most common circumstance was “separation from caregiver/parent.” Subjects had elevated scores on anxiety, irritability and hyperactivity scales with lower mean scores among subjects with a maternal deletion. The Pediatric Anxiety Rating Scale best captured behaviors described by caregivers. Existing anxiety scales should be adapted for use in AS.

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Acknowledgments

We would like to acknowledge the Angelman Syndrome Foundation for funding this study as well as the subjects and their family for their participation.

Funding

Funding was provided by Angelman Syndrome Foundation.

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Authors

Contributions

CJK, JEM, LN, RLT, CJM, and CTR contributed to the study conception and design. Material preparation, data collection and analysis were performed by CJK, JEM, LN, KW, BW, HKS, GE and CTR. The first draft of the manuscript was written by CKK and CTR and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

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Correspondence to Christopher J. Keary.

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Keary, C.J., Mullett, J.E., Nowinski, L. et al. Parent Description of Anxiety in Angelman Syndrome. J Autism Dev Disord 52, 3612–3625 (2022). https://doi.org/10.1007/s10803-021-05238-8

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