Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study.
Manning, Katherine E
Ring, Howard A
Porges, Stephen W
Jennings, Sally R
Public Library of Science (PLoS)
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Manning, K. E., Beresford-Webb, J., Aman, L., Ring, H. A., Watson, P., Porges, S. W., Oliver, C., et al. (2019). Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study.. PloS one, 14 (12), e0223750. https://doi.org/10.1371/journal.pone.0223750
Temper outbursts are a severe problem for people with Prader-Willi Syndrome (PWS). Previous reports indicate that vagus nerve stimulation (VNS) may reduce maladaptive behaviour in neurodevelopmental disorders, including PWS. We systematically investigated the effectiveness of transcutaneous VNS (t-VNS) in PWS. Using a non-blind single case repeat measures modified ABA design, with participants as their own controls, t-VNS was evaluated in five individuals with PWS [three males; age 22-41 (M = 26.8)]. After a baseline phase, participants received four-hours of t-VNS daily for 12 months, followed by one month of daily t-VNS for two-hours. The primary outcome measure was the mean number of behavioural outbursts per day. Secondary outcomes included findings from behavioural questionnaires and both qualitative and goal attainment interviews. Four of the five participants who completed the study exhibited a statistically significant reduction in number and severity of temper outbursts after approximately nine months of daily four-hour t-VNS. Subsequent two-hour daily t-VNS was associated with increased outbursts for all participants, two reaching significance. Questionnaire and interview data supported these findings, the latter indicating potential mechanisms of action. No serious safety issues were reported. t-VNS is an effective, novel and safe intervention for chronic temper outbursts in PWS. We propose these changes are mediated through vagal projections and their effects both centrally and on the functioning of the parasympathetic nervous system. These findings challenge our present biopsychosocial understanding of such behaviours suggesting that there is a single major mechanism that is modifiable using t-VNS. This intervention is potentially generalizable across other clinical groups. Future research should address the lack of a sham condition in this study along with the prevelance of high drop out rates, and the potential effects of different stimulation intensities, frequencies and pulse widths.
Vagus Nerve, Humans, Prader-Willi Syndrome, Treatment Outcome, Transcutaneous Electric Nerve Stimulation, Anger, Temperament, Adult, Female, Male, Vagus Nerve Stimulation, Self Report, Surveys and Questionnaires
Foundation for Prader-Willi Research (FPWR). Additional support came from the Prader-Willi Syndrome Association UK and Sam’s Foundation. The research was also facilitated by the National Institute for Health Research (NIHR) Collaboration for Leadership in Applied Health Research and Care (CLAHRC) East of England at Cambridgeshire and Peterborough NHS Foundation Trust. AJH was also supported by the Health Foundation.
Foundation for Prader-Willi Research (unknown)
Sam�s Research Foundation (via Prism the Gift Fund) (unknown)
External DOI: https://doi.org/10.1371/journal.pone.0223750
This record's URL: https://www.repository.cam.ac.uk/handle/1810/299939
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