Critical Review| Volume 14, ISSUE 9, P885-896, September 2013

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Behavioral Pain Indicators in People With Intellectual Disabilities: A Systematic Review


      People with intellectual disabilities (IDs) have a higher risk of painful medical conditions. Partly because of the impaired ability to communicate about it, pain is often undertreated. To strengthen pain assessment in this population, we conducted a systematic review to identify behavioral pain indicators in people with IDs by using Embase, PubMed, PsycINFO, CINAHL, and Cochrane. Inclusion criteria were 1) scientific papers; 2) published in the last 20 years, that is, 1992 to 2012; 3) written in English, 4) using human subjects, 5) intellectual disabilities, 6) pain, 7) behavior, and 8) an association between observable behavior and pain experience. From 527 publications, 27 studies were included. Pain was acute in 14 studies, chronic in 2 studies, both acute and chronic in 2 studies, and unspecified in 9 studies. Methodological quality was assessed with the Mixed Methods Appraisal Tool. Of the 14 categories with behavioral pain indicators, motor activity, facial activity, social-emotional indicators, and nonverbal vocal expression were the most frequently reported. Most of the behavioral pain indicators are reported in more than 1 study and form a possible clinical relevant set of indicators for pain in people with IDs. Determination of a behavioral pattern specific for pain, however, remains a challenge for future research.


      This review focuses on categories of behavior indicators related to pain in people with IDs. The quality of evidence is critically discussed per category. This set of indicators could potentially help clinicians to recognize pain in this population, especially when unique individual pain responses are also identified.

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        • American Psychiatric Association
        Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition Text Revision (DSM-IV-TR).
        American Psychiatric Association, Washington, DC2000
        • Baldridge K.H.
        • Andrasik F.
        Pain assessment in people with intellectual or developmental disabilities.
        Am J Nurs. 2010; 110: 28-35
        • Böhmer C.J.M.
        • Klinkenberg-Knol E.C.
        • Niezen-de Boer M.C.
        • Meuwissen S.G.M.
        Gastroesophageal reflux disease in intellectually disabled individuals: How often, how serious, how manageable?.
        Am J Gastroenterol. 2000; 95: 1868-1872
        • Bosch J.
        • Van Dyke D.C.
        • Smith S.M.
        • Poulton S.
        Role of medical conditions in the exacerbation of self-injurious behavior: An exploratory study.
        Ment Retard. 1997; 35: 124-130
        • Breau L.M.
        • Camfield C.S.
        The effects of psychopathology on the pain expression of children and youth with intellectual and developmental disabilities.
        J Ment Health Res Intellect Disabil. 2011; 4: 290-309
        • Breau L.M.
        • Camfield C.S.
        • McGrath P.J.
        • Finley G.A.
        The incidence of pain in children with severe cognitive impairments.
        Arch Pediatr Adolesc Med. 2003; 157: 1219-1226
        • Breau L.M.
        • Camfield C.S.
        • McGrath P.J.
        • Finley G.A.
        Pain's impact on adaptive functioning.
        J Intellect Disabil Res. 2007; 51: 125-134
        • Breau L.M.
        • Finley G.A.
        • McGrath P.J.
        • Camfield C.S.
        Validation of the Non-communicating Children's Pain Checklist–Postoperative Version.
        Anesthesiology. 2002; 96: 528-535
        • Breau L.M.
        • McGrath P.J.
        • Cainfield C.
        • Rosmus C.
        • Finley G.A.
        Preliminary validation of an observational pain checklist for persons with cognitive impairments and inability to communicate verbally.
        Dev Med Child Neurol. 2000; 42: 609-616
        • Burkitt C.C.
        • Breau L.M.
        • Zabalia M.
        Parental assessment of pain coping in individuals with intellectual and developmental disabilities.
        Res Dev Disabil. 2011; 32: 1564-1571
        • Carr E.G.
        • Smith C.E.
        • Giacin T.A.
        • Whelan B.M.
        • Pancari J.
        Menstrual discomfort as a biological setting event for severe problem behavior: Assessment and intervention.
        Am J Ment Retard. 2003; 108: 117-133
        • Collignon P.
        • Giusiano B.
        Validation of a pain evaluation scale for patients with severe cerebral palsy.
        Eur J Pain. 2001; 5: 433-442
        • Davies R.B.
        Pain in children with Down syndrome: Assessment and intervention by parents.
        Pain Manag Nurs. 2010; 11: 259-267
        • De Knegt N.
        • Scherder E.
        Pain in adults with intellectual disabilities.
        Pain. 2011; 152: 971-974
        • Dubois A.
        • Capdevila X.
        • Bringuier S.
        • Pry R.
        Pain expression in children with an intellectual disability.
        Eur J Pain. 2010; 14: 654-660
        • Espie C.A.
        • Watkins J.
        • Curtice L.
        • Espie A.
        • Duncan R.
        • Ryan J.A.
        • Brodie M.J.
        • Mantala K.
        • Sterrick M.
        Psychopathology in people with epilepsy and intellectual disability: An investigation of potential explanatory variables.
        J Neurol Neurosurg Psychiatry. 2003; 74: 1485-1492
        • Gibson S.J.
        Older people's pain.
        Pain: Clinical Updates. 2006; 14: 1-4
        • Giusiano B.
        • Jimeno M.T.
        • Collignon P.
        • Chau Y.
        Utilization of neural network in the elaboration of an evaluation scale for pain in cerebral palsy.
        Methods Inf Med. 1995; 34: 498-502
        • Herr K.
        • Coyne P.J.
        • Manworren R.
        • McCaffery M.
        • Merkel S.
        • Pelosi-Kelly J.
        • Wild L.
        Pain assessment in the nonverbal patient: Position statement with clinical practice recommendations.
        Pain Manag Nurs. 2006; 7: 44-52
        • Janicki M.P.
        • Davidson P.W.
        • Henderson C.M.
        • McCallion P.
        • Taets J.D.
        • Force L.T.
        • Sulkes S.B.
        • Frangenberg E.
        • Ladrigan P.M.
        Health characteristics and health services utilization in older adults with intellectual disability living in community residences.
        J Intellect Disabil Res. 2002; 46: 287-298
        • Kunz M.
        • Scharmann S.
        • Hemmeter U.
        • Schepelmann K.
        • Lautenbacher S.
        The facial expression of pain in patients with dementia.
        Pain. 2007; 133: 221-228
        • Kyrkou M.
        Health issues and quality of life in women with intellectual disability.
        J Intellect Disabil Res. 2005; 49: 770-772
        • LaChapelle D.L.
        • Hadjistavropoulos T.
        • Kenneth D.C.
        Pain measurement in persons with intellectual disabilities.
        Clin J Pain. 1999; 15: 13-23
        • Lotan M.
        • Ljunggren E.A.
        • Johnsen T.B.
        • Defrin R.
        • Pick C.G.
        • Strand L.I.
        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. 2009; 10: 398-407
        • Lotan M.
        • Moe-Nilssen R.
        • Ljunggren A.
        • Strand L.
        Measurement properties of the Non-Communicating Adult Pain Checklist (NCAPC): A pain scale for adults with intellectual and developmental disabilities, scored in a clinical setting.
        Res Dev Disabil. 2010; 31: 367-375
        • McCall W.V.
        Exacerbation of mental illness by dental disease.
        Psychosomatics. 1991; 32: 114-115
        • McGrath P.J.
        • Rosmus C.
        • Canfield C.
        • Campbell M.A.
        • Hennigar A.
        Behaviours caregivers use to determine pain in non-verbal, cognitively impaired individuals.
        Dev Med Child Neurol. 1998; 40: 340-343
        • McGuire B.E.
        • Daly P.
        • Smith F.
        Chronic pain in people with an intellectual disability: Under-recognised and under-treated?.
        J Intellect Disabil Res. 2010; 54: 240-245
      1. Merskey H. Bogduk N. Classification of Chronic Pain: Descriptions of Chronic Pain Syndromes and Definitions of Pain Terms. 2nd ed. IASP Press, Seattle, DC1994
        • Moher D.
        • Liberati A.
        • Tetzlaff J.
        • Altman D.G.
        • The PRISMA Group
        Preferred reporting items for systematic reviews and meta-analyses: The PRISMA statement.
        J Clin Epidemiol. 2009; 62: 1006-1012
        • Oberlander T.F.
        • Gilbert C.A.
        • Chambers C.T.
        • O'Donnell M.E.
        • Craig K.D.
        Biobehavioral responses to acute pain in adolescents with a significant neurological impairment.
        Clin J Pain. 1999; 15: 201-209
        • Patja K.
        • Iivanainen M.
        • Vesala H.
        • Oksanen H.
        • Ruoppila I.
        Life expectancy of people with intellectual disability: A 35-year follow-up study.
        J Intellect Disabil Res. 2000; 44: 591-599
        • Phan A.
        • Edwards C.L.
        • Robinson E.L.
        The assessment of pain and discomfort in individuals with mental retardation.
        Res Devel Disabil. 2005; 26: 433-439
      2. Pluye P, Robert E, Cargo M, Bartlett G, O'Cathain A, Griffiths F, Boardman F, Gagnon MP, Rousseau MC: Proposal: A mixed methods appraisal tool for systematic mixed studies reviews. 2011. Archived by WebCite® at Available at: Accessed October 9, 2012

        • Quint E.H.
        Menstrual issues in adolescents with physical and developmental disabilities.
        Ann N Y Acad Sci. 2008; 1135: 230-236
        • Quint E.H.
        • Elkins T.E.
        • Sorg C.A.
        • Kope S.
        The treatment of cyclical behavioral changes in women with mental disabilities.
        J Pediatr Adolesc Gynecol. 1999; 12: 139-142
        • Sandman C.A.
        • Hetrick W.
        • Taylor D.V.
        • Chicz-DeMet A.
        Dissociation of POMC peptides after self-injury predicts responses to centrally acting opiate blockers.
        Am J Ment Retard. 1997; 102: 182-199
        • Schneider M.J.
        • Bijam-Schulte A.M.
        • Janssen C.G.C.
        • Stolk J.
        The origin of self-injurious behaviour of children with mental retardation.
        Br J Dev Disabil. 1996; 42: 136-148
        • Shega J.W.
        • Rudy T.
        • Keefe F.J.
        • Perri L.C.
        • Mengin O.T.
        • Weiner D.K.
        Validity of pain behaviours in persons with mild to moderate cognitive impairment.
        J Am Geriatr Soc. 2008; 56: 1631-1637
        • Solodiuk J.C.
        • Scott-Sutherland J.
        • Meyers M.
        • Myette B.
        • Shusterman C.
        • Karian V.E.
        • Harris S.K.
        • Curley M.A.
        Validation of the Individualized Numeric Rating Scale (INRS): A pain assessment tool for nonverbal children with intellectual disability.
        Pain. 2010; 150: 231-236
        • Stein M.T.
        • Gottsegen D.
        • Blasco P.A.
        • Wolraich M.
        • Hennessy M.J.
        Chronic hip pain in a boy with mental retardation and cerebral palsy.
        J Dev Behav Pediatr. 2010; 31: S86-S91
        • Symons F.J.
        • Harper V.N.
        • McGrath P.J.
        • Breau L.M.
        • Bodfish J.W.
        Evidence of increased non-verbal behavioral signs of pain in adults with neurodevelopmental disorders and chronic self-injury.
        Res Dev Disabil. 2009; 30: 521-528
        • Symons F.J.
        • Harper V.N.
        • Shinde S.K.
        • Clary J.
        • Bodfish J.W.
        Evaluating a sham-controlled sensory-testing protocol for nonverbal adults with neurodevelopmental disorders: Self-injury and gender effects.
        J Pain. 2010; 11: 773-781
        • Symons F.J.
        • Shinde S.K.
        • Gilles E.
        Perspectives on pain and intellectual disabilities.
        J Intellect Disabil Res. 2008; 52: 275-286
        • Traci M.A.
        • Seekins T.
        • Szalda-Petree A.
        • Ravesloot C.
        Assessing secondary conditions among adults with developmental disabilities: A preliminary study.
        Ment retard. 2002; 40: 119-131
        • Tuffrey-Wijne I.
        • McEnhill L.
        Communication difficulties and intellectual disability in end-of- life care.
        Int J Palliat Nurs. 2008; 14: 189-194
        • Turk M.A.
        • Geremski C.A.
        • Rosenbaum P.F.
        • Weber R.J.
        The health status of women with cerebral palsy.
        Arch Phys Med Rehabil. 1997; 78: 10-17
        • Valkenburg A.J.
        • Boerlage A.A.
        • Ista E.
        • Duivenvoorden H.J.
        • Tibboel D.
        • van Dijk M.
        The COMFORT-behavior scale is useful to assess pain and distress in 0- to 3-year-old children with Down syndrome.
        Pain. 2011; 152: 2059-2064
        • Van der Putten A.
        • Vlaskamp C.
        Pain assessment in people with profound intellectual and multiple disabilities; a pilot study into the use of the Pain Behaviour Checklist in everyday practice.
        Res Dev Disabil. 2011; 32: 1677-1684
        • Vonken M.T.H.
        • Maaskant M.A.
        • Van den Akker M.
        Aandoeningen van het bewegingsapparaat bij mensen met een verstandelijke handicap.
        Nederlands Tijdschrift voor de Zorg aan verstandelijk gehandicapten (Dutch Journal for Care to Persons with Intellectual Disabilities). 2006; 32 ([Disorders of the movement system of people with intellectual disability]): 98-111
        • Walsh M.
        • Morrisson T.G.
        • McGuire B.E.
        Chronic pain in adults with an intellectual disability: Prevalence, impact, and health service use based on caregiver report.
        Pain. 2011; 152: 1951-1957
        • Zwakhalen S.M.G.
        • Van Dongen K.A.J.
        • Hamers J.P.H.
        • Abu-Saad H.H.
        Pain assessment in intellectually disabled people: Non-verbal indicators.
        J Adv Nurs. 2004; 45: 236-245
        • Zwakhalen S.M.G.
        • Hamers J.P.H.
        • Abu-Saad H.H.
        • Berger M.P.F.
        Pain in elderly people with severe dementia: A systematic review of behavioural pain assessment tools.
        BMC Geriatr. 2006; 6: 1-15