{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,3]],"date-time":"2026-11-03T21:23:51Z","timestamp":1793741031203,"version":"4.6.1"},"reference-count":40,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,3,19]],"date-time":"2021-03-19T00:00:00Z","timestamp":1616112000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2021,3,19]],"date-time":"2021-03-19T00:00:00Z","timestamp":1616112000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100005416","name":"Norges Forskningsr\u00e5d","doi-asserted-by":"publisher","award":["269348"],"award-info":[{"award-number":["269348"]}],"id":[{"id":"10.13039\/501100005416","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Sci Rep"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Intellectual disability (ID) affects approximately 1% of the population. Some patients with severe or profound ID are essentially non-communicating and therefore risk experiencing pain and distress without being able to notify their caregivers, which is a major health issue. This real-world proof of concept study aimed to see if heart rate (HR) monitoring could reveal whether non-communicating persons with ID experience acute pain or distress in their daily lives. We monitored HR in 14 non-communicating participants with ID in their daily environment to see if specific situations were associated with increased HR. We defined increased HR as being\u2009&gt;\u20091 standard deviation above the daily mean and lasting\u2009&gt;\u20095\u00a0s. In 11 out of 14 participants, increased HR indicated pain or distress in situations that were not previously suspected to be stressful, e.g. passive stretching of spastic limbs or being transported in patient lifts. Increased HR suggesting joy was detected in three participants (during car rides, movies). In some situations that were previously suspected to be stressful, absence of HR increase suggested absence of pain or distress. We conclude that HR monitoring may identify acute pain and distress in non-communicating persons with ID, allowing for improved health care for this patient group.<\/jats:p>","DOI":"10.1038\/s41598-021-86023-6","type":"journal-article","created":{"date-parts":[[2021,3,19]],"date-time":"2021-03-19T09:04:20Z","timestamp":1616144660000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":19,"title":["Increased heart rate functions as a signal of acute distress in non-communicating persons with intellectual disability"],"prefix":"10.1038","volume":"11","author":[{"given":"Emilie","family":"Kildal","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristine","family":"Stadskleiv","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elin S.","family":"Boysen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tone","family":"\u00d8derud","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Inger-Lise","family":"Dahl","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Trine M.","family":"Seeberg","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Svein","family":"Guldal","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frode","family":"Strisland","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cecilie","family":"Morland","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bj\u00f8rnar","family":"Hassel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,3,19]]},"reference":[{"key":"86023_CR1","doi-asserted-by":"publisher","first-page":"e20190811","DOI":"10.1542\/peds.2019-0811","volume":"144","author":"B Zablotsky","year":"2019","unstructured":"Zablotsky, B. et al. Prevalence and trends of developmental disabilities among children in the United States: 2009\u20132017. Pediatrics 144, e20190811 (2019).","journal-title":"Pediatrics"},{"key":"86023_CR2","doi-asserted-by":"publisher","first-page":"419","DOI":"10.1016\/j.ridd.2010.12.018","volume":"32","author":"PK Maulik","year":"2011","unstructured":"Maulik, P. K., Mascarenhas, M. N., Mathers, C. D., Dua, T. & Saxena, S. Prevalence of intellectual disability: A meta-analysis of population-based studies. Res. Dev. Disabil. 32, 419\u2013436 (2011).","journal-title":"Res. Dev. Disabil."},{"key":"86023_CR3","doi-asserted-by":"publisher","first-page":"80","DOI":"10.1016\/j.earlhumdev.2018.02.007","volume":"120","author":"JM Conway","year":"2018","unstructured":"Conway, J. M., Walsh, B. H., Boylan, G. B. & Murray, D. M. Mild hypoxic ischaemic encephalopathy and long term neurodevelopmental outcome\u2014A systematic review. Early Hum. Dev. 120, 80\u201387 (2018).","journal-title":"Early Hum. Dev."},{"key":"86023_CR4","doi-asserted-by":"publisher","first-page":"403","DOI":"10.1161\/STROKEAHA.107.489831","volume":"39","author":"D Ricci","year":"2008","unstructured":"Ricci, D. et al. Cognitive outcome at early school age in term-born children with perinatally acquired middle cerebral artery territory infarction. Stroke 39, 403\u2013410 (2008).","journal-title":"Stroke"},{"key":"86023_CR5","doi-asserted-by":"publisher","first-page":"509","DOI":"10.1016\/j.cortex.2019.07.020","volume":"120","author":"C de Montferrand","year":"2019","unstructured":"de Montferrand, C. et al. Language and cognitive outcomes after childhood stroke: Theoretical implications for hemispheric specialization. Cortex 120, 509\u2013523 (2019).","journal-title":"Cortex"},{"key":"86023_CR6","doi-asserted-by":"publisher","first-page":"133","DOI":"10.1016\/B978-0-444-64029-1.00006-0","volume":"162","author":"B Ostrander","year":"2019","unstructured":"Ostrander, B. & Bale, J. F. Congenital and perinatal infections. Handb. Clin. Neurol. 162, 133\u2013153 (2019).","journal-title":"Handb. Clin. Neurol."},{"key":"86023_CR7","doi-asserted-by":"publisher","first-page":"1161","DOI":"10.1126\/science.aar6731","volume":"362","author":"HC Martin","year":"2018","unstructured":"Martin, H. C. et al. Quantifying the contribution of recessive coding variation to developmental disorders. Science 362, 1161\u20131164 (2018).","journal-title":"Science"},{"key":"86023_CR8","doi-asserted-by":"publisher","DOI":"10.1176\/appi.books.9780890425596","volume-title":"Diagnostic and Statistical manual of Mental Disorders","author":"American Psychiatric Association","year":"2013","unstructured":"American Psychiatric Association. Diagnostic and Statistical manual of Mental Disorders 5th edn. (American Psychiatric Association, 2013).","edition":"5"},{"key":"86023_CR9","unstructured":"Carter, S. & Low, N.L.L. Cerebral palsy and mental retardation. In Merritt's Neurology, 13th ed. (eds. Louis, E.D., Mayer, S.A. & Rowland, L.P.) 1193\u20131194 (Wolters Kluwer, 2016)."},{"key":"86023_CR10","volume-title":"International Classification of Diseases 11th Revision","author":"World Health Organization","year":"2018","unstructured":"World Health Organization. International Classification of Diseases 11th Revision (World Health Organization, 2018)."},{"key":"86023_CR11","doi-asserted-by":"publisher","first-page":"361","DOI":"10.1111\/dmcn.12281","volume":"56","author":"M van Bakel","year":"2014","unstructured":"van Bakel, M. et al. Monitoring the prevalence of severe intellectual disability in children across Europe: Feasibility of a common database. Dev. Med. Child Neurol. 56, 361\u2013369 (2014).","journal-title":"Dev. Med. Child Neurol."},{"key":"86023_CR12","unstructured":"National Academies of Sciences, Engineering, and Medicine. Mental Disorders and Disabilities Among Low-Income Children. (eds. Boat, T.F. & Wu, J.T.) Chapter 9 (The National Academies Press, 2015)."},{"key":"86023_CR13","doi-asserted-by":"publisher","first-page":"727","DOI":"10.1007\/s00198-016-3740-2","volume":"28","author":"R Balogh","year":"2017","unstructured":"Balogh, R. et al. Low-trauma fractures and bone mineral density testing in adults with and without intellectual and developmental disabilities: A population study. Osteoporos. Int. 28, 727\u2013732 (2017).","journal-title":"Osteoporos. Int."},{"key":"86023_CR14","doi-asserted-by":"publisher","DOI":"10.1007\/s10803-020-04774-z","author":"EC Kurtz-Nelson","year":"2020","unstructured":"Kurtz-Nelson, E. C. et al. Brief report: Associations between self-injurious behaviors and abdominal pain among individuals with ASD-associated disruptive mutations. J. Autism Dev. Disord. https:\/\/doi.org\/10.1007\/s10803-020-04774-z (2020).","journal-title":"J. Autism Dev. Disord."},{"key":"86023_CR15","doi-asserted-by":"publisher","first-page":"e1002","DOI":"10.1542\/peds.2017-1002","volume":"139","author":"J Hauer","year":"2017","unstructured":"Hauer, J. & Houtrow, A. J. Section on hospice and palliative medicine, council on children with disabilities. Pain assessment and treatment in children with significant impairment of the central nervous system. Pediatrics 139, e1002 (2017).","journal-title":"Pediatrics"},{"key":"86023_CR16","doi-asserted-by":"publisher","first-page":"58","DOI":"10.1016\/j.pain.2003.12.003","volume":"108","author":"R Defrin","year":"2004","unstructured":"Defrin, R., Pick, C. G., Peretz, C. & Carmeli, E. A quantitative somatosensory testing of pain threshold in individuals with mental retardation. Pain 108, 58\u201366 (2004).","journal-title":"Pain"},{"key":"86023_CR17","doi-asserted-by":"publisher","first-page":"2399","DOI":"10.1016\/j.ridd.2013.04.011","volume":"34","author":"AA Boerlage","year":"2013","unstructured":"Boerlage, A. A. et al. Prevalence of pain in institutionalized adults with intellectual disabilities: A cross-sectional approach. Res. Dev. Disabil. 34, 2399\u20132406 (2013).","journal-title":"Res. Dev. Disabil."},{"key":"86023_CR18","doi-asserted-by":"publisher","first-page":"194","DOI":"10.3389\/fnbeh.2015.00194","volume":"9","author":"BE McGuire","year":"2015","unstructured":"McGuire, B. E. & Defrin, R. Pain perception in people with Down syndrome: A synthesis of clinical and experimental research. Front. Behav. Neurosci. 9, 194 (2015).","journal-title":"Front. Behav. Neurosci."},{"key":"86023_CR19","doi-asserted-by":"publisher","first-page":"441","DOI":"10.1093\/pm\/pnw349","volume":"18","author":"T Benromano","year":"2017","unstructured":"Benromano, T., Pick, C. G., Merick, J. & Defrin, R. Physiological and behavioral responses to calibrated noxious stimuli among individuals with cerebral palsy and intellectual disability. Pain Med. 18, 441\u2013453 (2017).","journal-title":"Pain Med."},{"issue":"2","key":"86023_CR20","doi-asserted-by":"publisher","first-page":"167","DOI":"10.1136\/archdischild-2019-318695","volume":"106","author":"S Bembich","year":"2021","unstructured":"Bembich, S. et al. Venipuncture activates the cerebral cortex in children with intellectual disability. Arch. Dis. Childhood 106(2), 167\u2013172 (2021).","journal-title":"Arch. Dis. Childhood"},{"key":"86023_CR21","doi-asserted-by":"publisher","first-page":"885","DOI":"10.1016\/j.jpain.2013.04.016","volume":"14","author":"NC De Knegt","year":"2013","unstructured":"De Knegt, N. C. et al. Behavioural pain indicators in people with intellectual disabilities: A systematic review. J. Pain 14, 885\u2013896 (2013).","journal-title":"J. Pain"},{"key":"86023_CR22","first-page":"824125","volume":"2014","author":"M Shanmugam","year":"2014","unstructured":"Shanmugam, M. et al. Behavioral pattern during dental pain in intellectually disabled children: A comparative study. Int. Sch. Res. Notices 2014, 824125 (2014).","journal-title":"Int. Sch. Res. Notices"},{"key":"86023_CR23","doi-asserted-by":"publisher","first-page":"129","DOI":"10.1111\/jpc.14230","volume":"55","author":"M Cascella","year":"2019","unstructured":"Cascella, M., Bimonte, S., Saettini, F. & Muzio, M. R. The challenge of pain assessment in children with cognitive disabilities: Features and clinical applicability of different observational tools. J. Paediatr. Child Health 55, 129\u2013135 (2019).","journal-title":"J. Paediatr. Child Health"},{"key":"86023_CR24","doi-asserted-by":"publisher","first-page":"77","DOI":"10.1177\/152574010002100202","volume":"21","author":"J Sigafoos","year":"2000","unstructured":"Sigafoos, J. et al. Identifying potential communicative acts in children with developmental and physical disabilities. Comm. Dis. Quart. 21, 77\u201386 (2000).","journal-title":"Comm. Dis. Quart."},{"key":"86023_CR25","doi-asserted-by":"publisher","first-page":"116","DOI":"10.1155\/2009\/642352","volume":"14","author":"LM Breau","year":"2009","unstructured":"Breau, L. M. & Burkitt, C. Assessing pain in children with intellectual disabilities. Pain Res. Manag. 14, 116\u2013120 (2009).","journal-title":"Pain Res. Manag."},{"key":"86023_CR26","doi-asserted-by":"publisher","first-page":"398","DOI":"10.1016\/j.jpain.2008.09.006","volume":"10","author":"M Lotan","year":"2009","unstructured":"Lotan, M. et al. A modified version of the non-communicating children pain checklist-revised, adapted to adults with intellectual and developmental disabilities: Sensitivity to pain and internal consistency. J. Pain 10, 398\u2013407 (2009).","journal-title":"J. Pain"},{"key":"86023_CR27","doi-asserted-by":"publisher","first-page":"55","DOI":"10.2174\/1874434601004010055","volume":"4","author":"P Kankkunen","year":"2010","unstructured":"Kankkunen, P., J\u00e4nis, P. & Vehvil\u00e4inen-Julkunen, K. Pain assessment among non-communicating intellectually disabled people described by nursing staff. Open Nurs. J. 4, 55\u201359 (2010).","journal-title":"Open Nurs. J."},{"key":"86023_CR28","doi-asserted-by":"crossref","first-page":"2472","DOI":"10.1111\/jocn.14834","volume":"28","author":"I Dillane","year":"2019","unstructured":"Dillane, I. & Doody, O. Nursing people with intellectual disability and dementia experiencing pain: An integrative review. J. Clin. Nurs. 28, 2472\u20132485 (2019).","journal-title":"J. Clin. Nurs."},{"key":"86023_CR29","doi-asserted-by":"publisher","first-page":"692","DOI":"10.1111\/j.1469-8986.1974.tb01138.x","volume":"11","author":"ER Hilgard","year":"1974","unstructured":"Hilgard, E. R. et al. Heart rate changes in pain and hypnosis. Psychophysiology 11, 692\u2013702 (1974).","journal-title":"Psychophysiology"},{"key":"86023_CR30","doi-asserted-by":"publisher","first-page":"603","DOI":"10.1016\/j.ejpain.2005.09.003","volume":"10","author":"Y Tousignant-Laflamme","year":"2006","unstructured":"Tousignant-Laflamme, Y. & Marchand, S. Sex differences in cardiac and autonomic response to clinical and experimental pain in LBP patients. Eur. J. Pain 10, 603\u2013614 (2006).","journal-title":"Eur. J. Pain"},{"key":"86023_CR31","doi-asserted-by":"publisher","first-page":"433","DOI":"10.1097\/EJA.0b013e328343d524","volume":"28","author":"T Ledowski","year":"2011","unstructured":"Ledowski, T., Stein, J., Albus, S. & MacDonald, B. The influence of age and sex on the relationship between heart rate variability, haemodynamic variables and subjective measures of acute post-operative pain. Eur. J. Anaesthesiol. 28, 433\u2013437 (2011).","journal-title":"Eur. J. Anaesthesiol."},{"key":"86023_CR32","doi-asserted-by":"publisher","first-page":"69","DOI":"10.1016\/0140-6736(92)90394-I","volume":"340","author":"PJ Whorwell","year":"1992","unstructured":"Whorwell, P. J., Houghton, L. A., Taylor, E. E. & Maxton, D. G. Physiological effects of emotion: Assessment via hypnosis. Lancet 340, 69\u201372 (1992).","journal-title":"Lancet"},{"key":"86023_CR33","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1016\/S0301-0511(00)00065-X","volume":"55","author":"SR Waldstein","year":"2000","unstructured":"Waldstein, S. R. et al. Frontal electrocortical and cardiovascular reactivity during happiness and anger. Biol. Psychol. 55, 3\u201323 (2000).","journal-title":"Biol. Psychol."},{"key":"86023_CR34","doi-asserted-by":"publisher","first-page":"974","DOI":"10.1197\/j.aem.2006.04.015","volume":"13","author":"CA Marco","year":"2006","unstructured":"Marco, C. A., Plewa, M. C., Buderer, N., Hymel, G. & Cooper, J. Self-reported pain scores in the emergency department: Lack of association with vital signs. Acad. Emerg. Med. 13, 974\u2013979 (2006).","journal-title":"Acad. Emerg. Med."},{"key":"86023_CR35","doi-asserted-by":"publisher","first-page":"19","DOI":"10.1016\/j.jemermed.2006.05.029","volume":"32","author":"P Bossart","year":"2007","unstructured":"Bossart, P., Fosnocht, D. & Swanson, E. Changes in heart rate do not correlate with changes in pain intensity in emergency department patients. J. Emerg. Med. 32, 19\u201322 (2007).","journal-title":"J. Emerg. Med."},{"key":"86023_CR36","doi-asserted-by":"publisher","first-page":"239","DOI":"10.1016\/j.jpain.2009.07.009","volume":"11","author":"S Oka","year":"2010","unstructured":"Oka, S. et al. Predictability of painful stimulation modulates subjective and physiological responses. J. Pain 11, 239\u2013246 (2010).","journal-title":"J. Pain"},{"key":"86023_CR37","doi-asserted-by":"publisher","first-page":"2191","DOI":"10.1001\/jama.2013.281053","volume":"310","author":"World Medical Association","year":"2013","unstructured":"World Medical Association. World Medical Association Declaration of Helsinki: Ethical principles for medical research involving human subjects. JAMA 310, 2191\u20132194 (2013).","journal-title":"JAMA"},{"key":"86023_CR38","volume-title":"Bayley Scales of Infant and Toddler Development (Bayley-III)","author":"N Bayley","year":"2009","unstructured":"Bayley, N. Bayley Scales of Infant and Toddler Development (Bayley-III) 3rd edn. (Pearson Assessment, 2009).","edition":"3"},{"key":"86023_CR39","volume-title":"Wechsler Preschool and Primary Scale of Intelligence (WPPSI-IV)","author":"D Wechsler","year":"2015","unstructured":"Wechsler, D. Wechsler Preschool and Primary Scale of Intelligence (WPPSI-IV) 4th edn. (Pearson Assessment, 2015).","edition":"4"},{"key":"86023_CR40","doi-asserted-by":"publisher","first-page":"1275416","DOI":"10.1080\/23311908.2016.1275416","volume":"4","author":"R Kurmanaviciute","year":"2017","unstructured":"Kurmanaviciute, R. & Stadskleiv, K. Assessment of verbal comprehension and non-verbal reasoning when standard response mode is challenging: A comparison of different response modes and an exploration of their clinical usefulness. Cogent. Psychol. 4, 1275416 (2017).","journal-title":"Cogent. Psychol."}],"container-title":["Scientific Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-86023-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-86023-6","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-86023-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,23]],"date-time":"2023-10-23T09:42:02Z","timestamp":1698054122000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.nature.com\/articles\/s41598-021-86023-6"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,3,19]]},"references-count":40,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2021,12]]}},"alternative-id":["86023"],"URL":"https:\/\/doi.org\/10.1038\/s41598-021-86023-6","relation":{"has-preprint":[{"id-type":"doi","id":"10.21203\/rs.3.rs-122056\/v1","asserted-by":"object"}]},"ISSN":["2045-2322"],"issn-type":[{"value":"2045-2322","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,3,19]]},"assertion":[{"value":"4 December 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"5 March 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"19 March 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The authors declare no competing interests.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"6479"}}