Novel insights into maladaptive behaviours in Prader–Willi syndrome: serendipitous findings from an open trial of vagus nerve stimulation
Tóm tắt
We 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.
Three 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.
Vagal 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.
We 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.
Từ khóa
Tài liệu tham khảo
Alonso‐Vanegas M. A., 2010, Chronic intermittent vagal nerve stimulation in the treatment of refractory epilepsy: experience in Mexico with 35 cases, Cirugia y Cirujanos, 78, 15
Bogacz A., 2003, Vagus nerve stimulation and weight loss [abstract], Epilepsia, 44, 125
Cukiert A., 2009, VNS in children with refractory generalized epilepsy or focal epilepsy due to diffuse cortical abnormalities or damage [abstract], Epilepsia, 50, 449
Holland A. J., 1993, Measurement of excessive appetite and metabolic changes in Prader–Willi syndrome, International Journal of Obesity Related Metabolic Disorders, 17, 527
Koren M. S., 2006, Vagus nerve stimulation does not lead to significant changes in body weight in patients with epilepsy, Epilepsy & Behavior, 19, 50
Rawlins P., 1997, Are there benefits, in addition to improved seizure control, for children receiving vagus nerve stimulation? [abstract], Epilepsia, 38, 155
Roslin M., 2003, Vagus Nerve Stimulation, 113
Schrander‐Stumpel C. Th. R. M., 2003, Prader–Willi syndrome: causes of death in an international series of 27 cases, American Journal of Medical Genetics A, 124, 333