{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,23]],"date-time":"2026-07-23T16:18:36Z","timestamp":1784823516104,"version":"3.55.0"},"reference-count":28,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2015,5,27]],"date-time":"2015-05-27T00:00:00Z","timestamp":1432684800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000377","name":"The Dunhill Medical Trust","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100000377","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Addenbrooke's Charitable Trust"},{"DOI":"10.13039\/501100004815","name":"Isaac Newton Trust","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100004815","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Prader-Willi Association UK"},{"name":"NIHR Collaborations for Leadership in Applied Health Care Research and Care (CLAHRC) East of England"},{"DOI":"10.13039\/501100000724","name":"Health Foundation","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100000724","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["J intellect Disabil Res"],"published-print":{"date-parts":[[2016,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>We report striking and unanticipated improvements in maladaptive behaviours in Prader\u2013Willi 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\u2013moderate intellectual disability (ID) and social and behavioural difficulties, alongside a characteristic and severe hyperphagia.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Three individuals with PWS underwent surgery to implant the VNS device. VNS was switched on 3\u2009months post\u2010implantation, with an initial 0.25\u2009mA output current incrementally increased to a maximum of 1.5\u2009mA as tolerated by each individual. Participants were followed up monthly.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>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\u2010seeking behaviour, but not weight, indicate that VNS may have potential as a novel treatment for such behaviours.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>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.<\/jats:p><\/jats:sec>","DOI":"10.1111\/jir.12203","type":"journal-article","created":{"date-parts":[[2015,5,28]],"date-time":"2015-05-28T13:35:21Z","timestamp":1432820121000},"page":"149-155","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":38,"title":["Novel insights into maladaptive behaviours in Prader\u2013Willi syndrome: serendipitous findings from an open trial of vagus nerve stimulation"],"prefix":"10.1111","volume":"60","author":[{"given":"K.\u2009E.","family":"Manning","sequence":"first","affiliation":[{"name":"Department of Psychiatry University of Cambridge  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.\u2009J.","family":"McAllister","sequence":"additional","affiliation":[{"name":"Department of Psychiatry University of Cambridge  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H.\u2009A.","family":"Ring","sequence":"additional","affiliation":[{"name":"Department of Psychiatry University of Cambridge  Cambridge UK"},{"name":"National Institute for Health Research (NIHR) Collaborations for Leadership in Applied Health Research and Care (CLAHRC) East of England  Cambridge UK"},{"name":"Cambridgeshire and Peterborough NHS Foundation Trust  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"N.","family":"Finer","sequence":"additional","affiliation":[{"name":"National Centre for Cardiovascular Prevention and Outcomes UCL Institute of Cardiovascular Sciences  London UK"},{"name":"Addenbrooke's Hospital  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.\u2009L.","family":"Kelly","sequence":"additional","affiliation":[{"name":"Department of Psychiatry University of Cambridge  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K.\u2009P.","family":"Sylvester","sequence":"additional","affiliation":[{"name":"Addenbrooke's Hospital  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P.\u2009C.","family":"Fletcher","sequence":"additional","affiliation":[{"name":"Department of Psychiatry University of Cambridge  Cambridge UK"},{"name":"Addenbrooke's Hospital  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"N.\u2009W.","family":"Morrell","sequence":"additional","affiliation":[{"name":"Addenbrooke's Hospital  Cambridge UK"},{"name":"Department of Medicine University of Cambridge  Cambridge UK"},{"name":"Papworth Hospital NHS Trust  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.\u2009R.","family":"Garnett","sequence":"additional","affiliation":[{"name":"Addenbrooke's Hospital  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.\u2009R.\u2009A.","family":"Manford","sequence":"additional","affiliation":[{"name":"Addenbrooke's Hospital  Cambridge UK"},{"name":"Bedford Hospital NHS Trust  Bedford UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.\u2009J.","family":"Holland","sequence":"additional","affiliation":[{"name":"Department of Psychiatry University of Cambridge  Cambridge UK"},{"name":"National Institute for Health Research (NIHR) Collaborations for Leadership in Applied Health Research and Care (CLAHRC) East of England  Cambridge UK"},{"name":"Cambridgeshire and Peterborough NHS Foundation Trust  Cambridge UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2015,5,27]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jocn.2007.07.083"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1080\/10874208.2012.650097"},{"key":"e_1_2_8_4_1","first-page":"15","article-title":"Chronic intermittent vagal nerve stimulation in the treatment of refractory epilepsy: experience in Mexico with 35 cases","volume":"78","author":"Alonso\u2010Vanegas M. A.","year":"2010","journal-title":"Cirugia y Cirujanos"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.appet.2006.07.080"},{"issue":"8","key":"e_1_2_8_6_1","first-page":"125","article-title":"Vagus nerve stimulation and weight loss [abstract]","volume":"44","author":"Bogacz A.","year":"2003","journal-title":"Epilepsia"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1191\/1478088706qp063oa"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.59.3.463"},{"issue":"11","key":"e_1_2_8_9_1","first-page":"449","article-title":"VNS in children with refractory generalized epilepsy or focal epilepsy due to diffuse cortical abnormalities or damage [abstract]","volume":"50","author":"Cukiert A.","year":"2009","journal-title":"Epilepsia"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1525-5050(03)00003-9"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.51.1.48"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ijo.0803128"},{"key":"e_1_2_8_13_1","first-page":"527","article-title":"Measurement of excessive appetite and metabolic changes in Prader\u2013Willi syndrome","volume":"17","author":"Holland A. J.","year":"1993","journal-title":"International Journal of Obesity Related Metabolic Disorders"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0033291702006736"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1177\/1049732305276687"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.yebeh.2010.06.012"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.yebeh.2003.12.008"},{"key":"e_1_2_8_18_1","first-page":"50","article-title":"Vagus nerve stimulation does not lead to significant changes in body weight in patients with epilepsy","volume":"19","author":"Koren M. S.","year":"2006","journal-title":"Epilepsy & Behavior"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.3171\/jns.1993.78.1.0026"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1528-1157.2003.56202.x"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ijo.0803666"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/icd.688"},{"issue":"8","key":"e_1_2_8_23_1","first-page":"155","article-title":"Are there benefits, in addition to improved seizure control, for children receiving vagus nerve stimulation? [abstract]","volume":"38","author":"Rawlins P.","year":"1997","journal-title":"Epilepsia"},{"key":"e_1_2_8_24_1","first-page":"113","volume-title":"Vagus Nerve Stimulation","author":"Roslin M.","year":"2003"},{"key":"e_1_2_8_25_1","first-page":"333","article-title":"Prader\u2013Willi syndrome: causes of death in an international series of 27 cases","volume":"124","author":"Schrander\u2010Stumpel C. Th. R. M.","year":"2003","journal-title":"American Journal of Medical Genetics A"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.88.3.263"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1097\/MPG.0b013e31805b82b5"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.38.11.792"},{"key":"e_1_2_8_29_1","doi-asserted-by":"crossref","first-page":"361","DOI":"10.1111\/j.1600-0447.1983.tb09716.x","article-title":"The hospital anxiety and depression scale","volume":"61","author":"Zigmond A. S.","year":"1983","journal-title":"Acta Psychiatrica et Neurologica Scandinavica"}],"container-title":["Journal of Intellectual Disability Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fjir.12203","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/jir.12203","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/jir.12203","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/jir.12203","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,14]],"date-time":"2023-09-14T05:21:16Z","timestamp":1694668876000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/jir.12203"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,5,27]]},"references-count":28,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2016,2]]}},"alternative-id":["10.1111\/jir.12203"],"URL":"https:\/\/doi.org\/10.1111\/jir.12203","archive":["Portico"],"relation":{},"ISSN":["0964-2633","1365-2788"],"issn-type":[{"value":"0964-2633","type":"print"},{"value":"1365-2788","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,5,27]]},"assertion":[{"value":"2014-11-24","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2015-04-14","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2015-05-27","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}