Evaluating a Sham-Controlled Sensory-Testing Protocol for Nonverbal Adults With Neurodevelopmental Disorders: Self-Injury and Gender Effects


      Ambiguous or blunted responses to sensory and painful stimuli among individuals with severe intellectual disabilities and comorbid communicative impairments put them at risk for having their experience of pain discounted and their expression of pain misinterpreted. Valid measurement procedures of behavioral expression are critical for this vulnerable group of individuals. We investigated a sham-controlled sensory-testing protocol as an approach to guard against observer bias during nonverbal behavioral recording for individuals with intellectual disabilities. Participants were 44 (52% male) adults (mean age = 46, sd = 10) with moderate (14%) and severe to profound (86%) intellectual impairment. The facial behavior of the participants before, during, and after 5 sensory-stimulation modalities (pin prick, light touch, deep pressure, cool, warm) was coded by 3 raters using the Facial Action Coding System (FACS). For each participant, the 5 active sensory trials were randomized with sham trials during which no stimulation was applied. Observers were blinded to active vs sham stimulation status. FACS scores increased significantly during active sensory trials (P < .05) compared with sham trials. There were significant effects for gender, with females more expressive than males (P < .05). There were also significant effects for the presence of self-injurious behavior (SIB), with individuals with SIB more expressive than individuals without SIB (P < .05). The results suggest that the procedure was valid (ie, distinguished between active vs sham sensory stimulation) and provides additional evidence that individuals with significant intellectual impairments are sensitive to tactile stimulation consistent with quantitative sensory-testing protocols.


      This article presents a novel application of a modified approach to quantitative sensory testing for nonverbal adults with intellectual and developmental disabilities. This approach could be important in helping determine sensory issues related to tactile and nociceptive processes among a highly vulnerable group of individuals.

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        • Abu-Saad H.H.
        Challenge of pain in the cognitively impaired.
        Lancet. 2000; 356: 1867-1868
        • Barrera F.J.
        • Teodoro J.M.
        • Selmeci T.
        • Madappuli A.
        Self-injury, pain, and the endorphin theory.
        J Dev Phys Dis. 1994; 6: 169-192
        • Biersdorff K.
        Pain insensitivity and indifference: Alternative explanations for some medical catastrophes.
        Ment Retard. 1991; 29: 359-362
        • Biersdorff K.
        Incidence of significantly altered pain experience among individuals with developmental disabilities.
        Am J Ment Retard. 1994; 98: 619-631
        • Breau L.M.
        • Camfield C.S.
        • Symons F.J.
        • Bodfish J.W.
        • MacKay A.
        • Finley G.A.
        • McGrath P.J.
        Relation between pain and self-injurious behavior in nonverbal children with severe cognitive impairments.
        J Ped. 2003; 142: 498-503
        • Breau L.M.
        • McGrath P.J.
        • Craig K.D.
        • Santor D.
        • Cassidy K.L.
        • Reid G.J.
        Facial expression of children receiving immunizations: A principle-components analysis of the Child Facial Coding System.
        Clin J Pain. 2001; 17: 178-186
        • Craig K.D.
        The facial display of pain.
        in: Finley G.A. McGrath P.J. Measurement of Pain in Infants and Children. International Association for the Study of Pain Press, Seattle, WA1998 (pp 103-122)
        • Craig K.D.
        • Prkachin K.M.
        • Grunau R.E.
        The facial expression of pain.
        in: Turk D.C. Melzack R. Handbook of Assessment of Pain. 2nd ed. The Guilford Press, New York, NY2001: 153-169
        • Defrin R.
        • Lotan M.
        • Pick C.G.
        The evaluation of acute pain in individuals with cognitive impairment: A differential effect of the level of impairment.
        Pain. 2006; 124: 312-320
        • Defrin R.
        • Pick C.G.
        • Peretz C.
        • Carmeli E.
        A quantitative somatosensory testing of pain threshold in individuals with mental retardation.
        Pain. 2004; 108: 58-66
        • Ekman P.
        • Friesen W.V.
        • Hager J.C.
        Facial Action Coding System: The manual.
        Research Nexus Division of Network Information Research Corporation, Salt Lake City, UT2002
        • Fillingim R.B.
        Sex, gender, and pain.
        Curr Rev Pain. 2000; 4: 24-30
        • Grunau R.V.
        • Craig K.D.
        Pain expression in neonates: Facial action and cry.
        Pain. 1987; 28: 395-410
        • Hadjistavropoulos T.
        • von Baeyer C.
        • Craig K.D.
        Pain assessment in persons with limited availability to communicate.
        in: Turk D.C. Melzack R. Handbook of Pain Assessment. 2nd ed. The Guilford Press, New York, NY2001 (pp 134-152)
        • Hennequin M.
        • Morin C.
        • Feine J.S.
        Pain expression and stimulus localization in individuals with Down syndrome.
        Lancet. 2000; 356: 1882-1887
        • Iwata B.A.
        • Pace G.M.
        • Kissel R.C.
        • Nau P.A.
        • Farber J.M.
        The self-injury trauma (SIT) scale: A method for quantifying surface tissue damage caused by self-injurious behavior.
        J App Beh Analy. 1990; 23: 99-110
        • LaChapelle D.L.
        • Hadjistavropoulos T.
        • Craig K.D.
        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's pain checklist-revised, adapted to adults with intellectual and developmental disabilities: Sensitivity to pain and internal consistency.
        J Pain. 2009; 10: 398-407
        • Nader R.
        • Oberlander T.F.
        • Chambers C.T.
        • Craig K.D.
        Expression of pain in children with autism.
        Clin J Pain. 2004; 20: 88-97
        • Oberlander T.F.
        • Craig K.D.
        Pain and children with developmental disabilities.
        in: Schechter N.L. Berde C.B. Yasters M. Pain in Infants, Children, and Adolescents. 2nd ed. Lippincott Williams & Williams, New York, NY2003: 599-619
        • 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 neurologic impairment.
        Clin J Pain. 1999; 15: 201-209
        • Oberlander T.F.
        • Symons F.
        Pain in children and adults with developmental disabilities.
        Paul H. Brookes Publishing Co, Baltimore, MD2006
        • Oberlander T.F.
        • Symons F.
        • van Dongen K.
        Abu-Saad HH: Pain in individuals with developmental disabilities: Challenges for the future.
        Progress in Pain Research and Management, Vol. 24. Proceedings of the 10th World Congress on Pain. International Association for the Study of Pain Press, Seattle, WA2003
        • Phan A.
        • Edwards C.L.
        • Robinson E.L.
        The assessment of pain and discomfort in individuals with mental retardation.
        Res Develop Disabil. 2005; 26: 433-439
        • Prkachin K.M.
        The consistency of facial expressions of pain: A comparison across modalities.
        Pain. 1992; 51: 297-306
        • Prkachin K.M.
        The coming of age of pain expression.
        Pain. 2007; 133: 3-4
        • Prkachin K.M.
        • Craig K.D.
        Expressing pain: The communication and interpretation of facial pain signals.
        J Nonverb Beh. 1995; 19: 191-205
        • Rojahn J.
        • Matson J.L.
        • Lott D.
        • Esbensen A.J.
        • Smalls Y.
        The Behavior Problems Inventory: An instrument for the assessment of self-injury, stereotyped behavior, and aggression/destruction in individuals with developmental disabilities.
        J Aut Dev Dis. 2001; 31: 577-588
        • Rolke R.
        • Magerl W.
        • Campbell K.A.
        • Schalber C.
        • Caspari S.
        • Birklein F.
        • Treede R.D.
        Quantitative sensory testing: A comprehensive protocol for clinical studies.
        E J Pain. 2005; 10: 77-88
        • Sandman C.A.
        The opiate hypothesis in autism and self-injury.
        J Child Adoles Psychopharm. 1991; 1: 237-248
        • Sandman C.A.
        • Spence M.A.
        • Smith M.
        Proopiomelanocortin (POMC) disregulation and response to opiate blockers.
        Ment Ret Develop Dis Res Rev. 1999; 5: 314-321
        • Sobsey D.
        Pain and disability in an ethical and social context.
        in: Oberlander T.F. Symons F.J. Pain in children and adults with developmental disabilities. Paul H. Brookes Publishing Co, Baltimore, MD2006: 19-39
        • Symons F.J.
        Pain and self-injury: Mechanisms and models.
        in: Schroeder S. Thompson T. Oster-Granite M.L. Self-Injurious Behavior: Genes, Brain, and Behavior. American Psychological Association, Washington, DC2002: 223-234
        • Symons F.J.
        • Danov S.
        A prospective clinical analysis of pain behavior and self-injurious behavior.
        Pain. 2005; 117: 473-477
        • Tapp J.
        Procoder for digital video user manual.
        The John F. Kennedy Center at Vanderbilt University, Nashville, TN2003
        • Wiesenfeld-Hallin Z.
        Sex differences in pain perception.
        Gend Med. 2005; 2: 137-145