Novel insights into maladaptive behaviours in Prader–Willi syndrome: serendipitous findings from an open trial of vagus nerve stimulation

Journal of Intellectual Disability Research - Tập 60 Số 2 - Trang 149-155 - 2016
Katherine E. Manning1, Catherine McAllister1, Howard Ring2,1,3, Nick Finer4,5, Claire Kelly1, Karl Sylvester4, Paul C. Fletcher4,1, Nicholas W. Morrell4,6,7, Mathew Garnett4, Mark Manford4,8, Anthony Holland2,1,3
1Department of Psychiatry, University of Cambridge, Cambridge, UK
2Cambridgeshire and Peterborough NHS Foundation Trust, Cambridge, UK
3National Institute for Health Research (NIHR) Collaborations for Leadership in Applied Health Research and Care (CLAHRC) East of England, Cambridge, UK
4Addenbrooke's Hospital, Cambridge, UK
5National Centre for Cardiovascular Prevention and Outcomes, UCL Institute of Cardiovascular Sciences, London, UK
6Department of Medicine, University of Cambridge, Cambridge, UK
7Papworth Hospital NHS Trust, Cambridge, UK
8Bedford Hospital NHS Trust, Bedford, UK

Tóm tắt

AbstractBackgroundWe report striking and unanticipated improvements in maladaptive behaviours in Prader–Willi syndrome (PWS) during a trial of vagus nerve stimulation (VNS) initially designed to investigate effects on the overeating behaviour. PWS is a genetically determined neurodevelopmental disorder associated with mild–moderate intellectual disability (ID) and social and behavioural difficulties, alongside a characteristic and severe hyperphagia.MethodsThree individuals with PWS underwent surgery to implant the VNS device. VNS was switched on 3 months post‐implantation, with an initial 0.25 mA output current incrementally increased to a maximum of 1.5 mA as tolerated by each individual. Participants were followed up monthly.ResultsVagal nerve stimulation in these individuals with PWS, within the stimulation parameters used here, was safe and acceptable. However, changes in eating behaviour were equivocal. Intriguingly, unanticipated, although consistent, beneficial effects were reported by two participants and their carers in maladaptive behaviour, temperament and social functioning. These improvements and associated effects on food‐seeking behaviour, but not weight, indicate that VNS may have potential as a novel treatment for such behaviours.ConclusionsWe propose that these changes are mediated through afferent and efferent vagal projections and their effects on specific neural networks and functioning of the autonomic nervous system and provide new insights into the mechanisms that underpin what are serious and common problems affecting people with IDs more generally.

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Journal of Intellectual Disability Research

Tập 60 Số 2

149-155

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