ABSTRACT
Background
Williams syndrome (WS) is a rare genetic disorder associated with a high prevalence of anxiety disorders. Evidence-based pharmacologic treatments for anxiety in WS are lacking. The purpose of this study is to provide naturalistic data on the use of buspirone for the treatment of anxiety in WS.
Research design and methods
Medical records of 24 individuals with Williams syndrome (ages 7–47 years) and anxiety who received treatment with buspirone were reviewed. Treatment response to buspirone was rated by assigning a retrospective Clinical Global Impression Improvement subscale (CGI-I) score.
Results
Twenty-three of 24 (96%) patients completed at least a 16-week treatment course with buspirone. Sixteen patients (67%; 95% CI 47%, 82%) were treatment responders (CGI-I ≤ 2). Only 1 (4%) patient discontinued buspirone due to a treatment-emergent side effect (nausea and vomiting). The most common side effect was nausea (13%). Twenty (84%) patients remained on buspirone at the time of their most recent follow-up visit.
Conclusions
In this retrospective study, the majority of patients responded to a 16-week course of buspirone. Prospective studies are warranted to further assess the efficacy and tolerability of buspirone for anxiety in WS.
Declaration of interest
R P Thom has received research funding from the Williams Syndrome Association. The authors have no other relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript apart from those disclosed.
Author contributions
E Shin was involved in drafting the manuscript. D Renzi was involved in study design and statistical analysis. C Canales was involved in data collection. C Ravichandran was involved in study design, statistical analysis, and revising the manuscript critically for intellectual content. C J McDougle was involved in study conception and design and revising the manuscript critically for intellectual content. R P Thom was involved in study conception and design, data collection, drafting the manuscript, and revising it critically for intellectual content. All authors have provided final approval of the version to be published and agree to be accountable for all aspects of the work.
Acknowledgments
The authors would also like to acknowledge the Bernon family’s support.
Reviewer disclosures
Peer reviewers on this manuscript have no relevant financial or other relationships to disclose.