Pre-post design; outcome was measured by questionnaires on physical (e.g. pain, sleep quali ty, fatigue) and psychological (e.g. anxiety, internalization, quality of life) symptoms co mpleted by parents and children 8 weekly sessions which focused on psycho-education about pain and sleep, sleep improvement , pain management, and activities of daily living. Sleep treatment strategies included slee p hygiene, relaxation, self-reward, stimulus control, sleep restriction, and cognitive rest ructuring. Treatment was based on a CBT treatment manual developed by the authors Improvements reported across all measures, including decreased pain, somatic symptoms, anxi ety and fatigue as well as improved sleep quality. 24% of the children were reported to be pain free at the end of the intervention. Both self-reports and clinical observations showe d dramatic improvements. Results were based on participants who completed the intervention (34% attrition rate) Maynard et al., 2010 N = 41 (30 females, 11 males); pain-associated disability syndrome; mean age: 13.8 years (S D = 2.8, range = 8 \200\22321) Retrospective chart review; outcome was measured by information in chart on school attendan ce, medication usage, sleep, functional disability, coping skills, and mobility Treatment through an interdisciplinary inpatient rehabilitation program including several c omponents. Psychological treatment focused on CBT for pain and anxiety and the use of shapi ng to improve functional ability. Sleep intervention included relaxation strategies at bedt ime Improvement in functional ability, physical mobility, as well as school status, sleep, and medication use Palermo et al., 2009 N = 48 (35 females, 13 males); chronic headache, abdominal, or musculoskeletal pain; mean a ge: 14.8 years (SD = 2.0, range = 11 \200\22317) RCT; Internet-treatment group (n = 26), waitlist control group (n = 22); outcomes (e.g. rec ording pain and activity levels) were measured by daily diaries and questionnaires pre- and post-treatment Internet-delivered CBT intervention which included 8 weeks of modules focused on treating p ain through relaxation training, cognitive strategies, parent operant techniques, communica tion strategies, and sleep and activity interventions. Sleep intervention included informat ion on sleep and good sleep hygiene practices Improvements in pain intensity and activity levels at end of treatment and 3-month follow-u p. No improvement in parental protectiveness or child mood symptoms Bruni et al., 1999 N = 70 (32 females, 38 males); migraine su \200 erers with poor sleep hygiene; mean age: 9 .7 years (SD = 1.5, range = 5 \200\22315 years) RCT, assigned to treatment (n = 35), no treatment (n = 35); outcomes were assessed by track ing duration, frequency and severity of migraines Treatment included application of sleep hygiene guidelines, but these were not speci \201 ed in the paper Improvement in frequency and duration of migraines for treatment group, but no change in se verity described in detail in Table 15.2 . Results across these studies were generally positive, w ith improvements in both pain and sleep, as well as other associated areas (e.g. functional disability, fatigue). A series of randomized trials conducted among adults with chronic pa in conditions (Currie et al., 2000; Edinger et al., 2005; Jungquist et al., 2010; Rybarczyk et al., 2005) has yielded similar positive results. More intervention research in this are a is needed, with a focus on demonstrating that improvements in sleep does indeed result in improved pain outcomes. of childhood insomnia. Of note, many pain medications that are prescribed oft en have sleep -related eff ects. For example, tricyclic antidepressants and anticonvulsants oft en have s leepiness as a side eff ect which may help promote sleep. Th ese same medications may also cause daytime sleepiness or fatigue which may further worsen functional adaptation in deali ng with pain. In some instances, such as with the use of gabapentin for neuropathic pain, t he medication may help reduce pain and possibly improve a comorbid sleep dis- order such as restless legs syndrome. Selective serotonin reuptake inhibitors (e.g. paroxetine) may be u sed for treatment of comorbid psychological disorders rather than pain itself and the impac t of these medications on sleep such as rapid eye movement suppres- sion, or increased peri odic limb movements (Vendrame et al., 2011) should not be overlooked. Similarly, opioid med ications for chronic pain may worsen underlying sleep disordered breathing, or result in co nstipation and associated discomfort that impairs sleep. Controlled trials using medication s for treating chronic pain in children and teens are lacking. Future research studies shou ld add in components that address sleep specifi cally. (For more infor- mation on clinical use of opioids see Strassels, Chapter 45, and for Pharmacological treatments Pharmacological therapy for chronic pain should take into con- s ideration potential interactions with sleep. Th ere are no approved medications for the tre atment of insomnia in children or medica- tions for chronic pain in children. Reviews about insomnia and treatment options in children are available (Owens and Moturi, 2009; Owens et al., 2005). Chronic pain in children is oft en managed with similar medications as in adul ts off label, but integration of behavioral strategies are oft en paramount, much like mana gement SECTION 3 social and psychological basis of pediatric pain 140 drugs for neuropathic pain, see Rastogi and Campbell, Chapter 48, this volume.) Box 15.1 Healthy sleep habits for children and adolescents \227\206 Ensure morning bright-light exposure Key recommendations Given the evidence to date, we recommend that children with pain and sl eep problems be provided with a developmentally appropri- ate, evidence-based intervention for their sleep problems as one component of their pain management plan. A detailed descrip tion of these intervention strategies is beyond the scope of this review; however, Box 15.1 lists some healthy sleep habits for children and adolescents and Table 15.3 contains a lis t of resources. Some modi- fi cations or special considerations might be useful to consider when implementing sleep intervention for children with chronic pain (Smith et al., 2005). Th e clinician may have to explain to parents why treating sleep problems is important, as a study by Claar and Scharff (2007) found that parents did not see sleep interventions as l ikely to help with the treatment of pain in their children. It will also be important to de velop sleep interventions that parents of children with chronic pain can readily access. Fo r example, novel delivery methods, such as internet (Corkum and Davidson, 2011) or tel- ehe alth (Witmans et al., 2008) may help to reduce access barriers. Clinicians working with chi ldren with chronic pain may also want to consider the following when implementing sleep int erventions: \227\206 Schedule daily physical activity (ideally earlier in the day) \227\206 Avoid caff eine in the aft ernoon and evening \227\206 Reduce (and avoid where possible) bright light in the evening and in the night \227\206 Reduce stimulating activities before and at bedtime \227\206 Set an age-appropriate bedtime \227\206 Establish a regular, consistent bedtime routine \227\206 Maintain a consistent schedule for weekdays and weekends \227\206 Create an environment conducive to sleep: cool, dark, quiet, comfortable \227\206 Remove electronics from the bedroom \227\206 Use the bed only for sleep \227\206 Monitor for side eff ects of medications that may aff ect sleep. Table 15.3 Resources for sleep intervention in children \227\206 Managing pain will help the child be able to fall asleep and stay asleep. Also, m aking sure that medications taken by the child are not interfering with sleep. Journal articles Hill, C. (2011). Practitioner review: e \200 ective treatment of behavioral insomnia in ch ildren. J Child Psychol Psychiatry , 52(7), 731 \200\223741. doi:10.1111/j.1469 \200\223761 0.2011.02396.x Jan, J. E., Owens, J. A., Weiss, M. D., Johnson, K. P., Wasdell, M. B., Free man, R. D., et al . (2008). Sleep hygiene for children with neurodevelopmental disabilities . Pediatrics , 122(6), 1343 \200\2231350. doi:10.1542/peds.2007 \200\2233308 Meltzer, L. J. (2010). Clinical management of behavioral insomnia of childhood: treatment of bedtime prob lems and night wakings in young children. Behav Sleep Med , 8(3), 172 \200\223189. doi:10.1 080/15402002.2010.487464 Taylor, D. J., and Roane, B. M. (2010). Treatment of insomnia in a dults and children: a practice-friendly review of research. J Clin Psychol , 66(11), 1137 \200\2231147. doi:10.1002/jclp.20733 Vriend, J. and Corkum, P. (2011). Clinical management of behavioral insomnia of childhood. Psychol Res Behav Manag , 4, 69 \200\22379. doi: http: //dx.doi.org/10.2147/PRBM.S14057 \227\206 Many of the strategies used to help with pain management (e.g. proper diet and ex ercise) will also help with sleep. It is important that exercise does not occur close to be dtime and that foods that cause any intestinal distress not be eaten close to bedtime. \227\206 Parents and children should be informed about the relationship between sleep and pain, so that they understand why it is impor- tant to treat sleep problems as part of the overall pain manage- ment plan. For example, it would be important to point out that poor s leep is associated with next-day pain as well as increased negative aff ect and decreased p ositive aff ect. \227\206 Sleep hygiene is an important component for sleep treatment. For children with ch ronic pain, it is important that the bed/bed- room is not associated with any painful treat ments (e.g. injec- tions) and that the child \200\231s bed is as comfortable as possible in order to reduce any pain associated with sleeping. Books for professionals Durand, V. M. (2008). When children don \200\231t sleep well: parent workbook: intervention s for pediatric sleep disorders (treatments that work). New York: Oxford University Press M indell, J. A. and Owens, J.A. (2009). A clinical guide to pediatric sleep: diagnosis and ma nagement of sleep problems (2nd edn). Philadelphia, PA: Lippincott Williams & Wilkins Sheld on, S. S., Ferber, R. and Kryger, M. H. (2005). Principles and practice of pediatric sleep medicine . Philadelphia, PA: W.B. Saunders Stores, G. and Wiggs, L. (2001). Sleep disturban ce in children and adolescents with disorders of development: its signi \201 cance and man agement . London: MacKeith Press \227\206 Given that children with pain may experience hyperarousal, it is important to inc lude relaxation training as one of the sleep inter- vention strategies. \227\206 Sleep restriction therapy should be used with caution given some evidence that sleep restriction can increase pain. \227\206 Parents may require additional support when implementing sleep strategies. Many p arents fi nd implementing these strategies to be stressful and this may be more so for pare nts of children with chronic pain. Websites Kidszzzsleep: <http://www.kidzzzsleep.org> National Sleep Foundation: <http://www.sleepfoun dation. org> Sleep Net: <http://www.sleepnet.com/> Sleep for Kids: <http://www.sleepforkids .org/index.html> Conclusion Th ere is much more to learn about which sleep treatment compo- nents are most e ff ective in this population, as well as how we might CHAPTER 15 sleep and pain in children and adolescents 141 have to modify existing sleep interventions to enhance eff ectiveness in children with chro nic pain. However, at the current time, it would seem reasonable for clinicians working wit h children with chronic pain to assess and treat sleep problems. At a minimum, treating sle ep problems will reduce the number of issues with which the child and parent is trying to c ope, and could potentially improve pain while enhancing overall quality of life. and disease activity in juvenile rheumatoid arthritis. J Rheumatol, 29 , 169 \200\223173 . Breau, L. M. and Camfi eld, C. S. (2011). Pain disrupts sleep in children and youth with in tellectual and developmental disabilities. Res Dev Disabil, 32, 2829 \200\2232840 . Bromberg, M. H., Gil, K. M., and Schanberg, L. E. (2011). Daily sleep quality and mood as p redictors of pain in children with juvenile polyarticular arthritis. Health Psychol, 31, 20 2 \200\223209 . Th ere is now a strong empirical base to conclude that sleep disturbances are indeed a sign ifi cant problem for children with chronic pain. Th ere remains much important work to do i n this area. For example, more research is needed to examine the develop- mental trajectory of the relationship between sleep and pain prob- lems across childhood and adolescence, th e impact of the kinds and quantities of medication used by children with chronic pain and h ow these relate to sleep (Chambers et al., 2008). Future research is critically needed to d evelop and test the impact of interventions addressing sleep and pain among children and ad olescents with chronic pain. Brooks, L. J., Koziol, S. M., Chiarucci, K. M., and Berman, B. W. (1996). Does sleep-disord ered breathing contribute to the clinical severity of sickle cell anemia? J Pediatr Hematol /Oncol, 18, 135 \200\223139 . Bruni, O., Galli, F., and Guidetti, V. (1999). Sleep hygiene and migraine in children and adolescents. Cephalalgia, 19, 57 \200\22359 . Butbul Aviel, Y., Stremler, R., Benseler, S. M., Cameron, B., Laxer, R. M., Ota, S., et al . (2011). Sleep and fatigue and the relationship to pain, disease activity and quality of l ife in juvenile idiopathic arthritis and juvenile dermatomyositis. Rheumatology, 50, 2051 \200\2232060 . Cappuccio, F. P., Taggart, F. M., Kandala, N. B., Currie, A., Peile, E., Stranges, S., et a l . (2008). Meta-analysis of short sleep duration and obesity in children and adults. Sleep , 31, 619 \200\223626 . Chambers, C. T., Corkum, P., and Rusak, B. (2008). Commentary: the importance of sleep in p ediatric chronic pain \200\224a wake-up call for pediatric psychologists. J Pediatr Psychol , 33, 333 \200\223334 . Case example: pain and sleep Claar, R. and Scharff , L. (2007). Parent and child perceptions of chronic pain Melissa is a 14-year-old girl with a 5-year history of recurrent abdominal pain, which had recently signifi cantly worsened to the point that she was now unable to attend school. At the time of her pain clinic evaluation, Melissa was experiencing daily, severe stom- ach ac hes with no identifi able medical cause. When queried about sleep during her evaluation, Me lissa noted that in addition to her pain she was experiencing signifi cant diffi culties fa lling asleep at night and would watch television in bed until late at night. Melissa was al so having great diffi culty getting out of bed at a reason- able time in the morning. Becau se she was not attending school, her parents tended to let Melissa sleep in to catch up on her sleep, and Melissa reported that she oft en napped in the aft ernoon while watching tel evision and resting. Melissa reported feeling more irri- table and fatigued during the day, and acknowledged that feeling this way was likely making it harder for her to deal with he r pain. Th e pain management team recommended a comprehensive cognitive- behavioral interve ntion in order to help Melissa learn strategies to better cope with her pain as well as a g raduated school re-entry plan. Melissa \200\231s sleep, in addition to her pain, was also i dentifi ed as one of the primary treatment targets. Th e family was provided with basic inf ormation about sleep and sleep hygiene and links between pain and sleep problems. A session was scheduled to tailor those strategies to meet Melissa \200\231s needs and to help her p arents coach and support her sleep changes. Th e importance of a regular sleep schedule, th e removal of the television from the bedroom, and the negative impact of daytime naps on ni ght sleep were discussed. As treatment progressed, Melissa noted signifi cant improvements in her sleep and her ability to apply the pain coping strategies she was learning in clinic . At the end of treatment, Melissa was still experiencing mild stomach aches most days, but had returned to school, was sleeping much better, her mood had improved, and she reported feeling more energy and less irritability. treatments. Child Health Care, 36, 285 \200\223301 . Corkum, P. and Davidson, F. (2011). Sleep intervention for behavioral insomnia in children: impact on sleep and daytime functioning of typically developing children and children with ADHD. Presentation at the AACAP/CACAP Joint Annual Meeting, 18 \200\22323 October, Toronto , Ontario . Currie, S. R., Wilson, K. G., Pontefract, A. J., and deLaplante, L. (2000). Cognitive \200 \223behavioral treatment of insomnia secondary to chronic pain. J Consult Clin Psych, 68, 4 07 \200\223416 . Degotardi, P. J., Klass, E. S., Rosenberg, B. S., Fox, D. G., Gallelli, K. A., and Gottlieb , B. S. (2006). Development and evaluation of a cognitive- behavioral intervention for juve nile fi bromyalgia. J Ped Psych , 31, 714 \200\223723 . Dick, B. D. and Pillai Riddell, R. (2010). Cognitive and school functioning in children and adolescents with chronic pain: a critical review. Pain Res Manag, 15, 238 \200\223244 . Durmer, J. S. and Dinges, D. F. (2005). Neurocognitive consequences of sleep deprivation. Semin Neurol , 25, 117 \200\223129 . Edinger, J. D., Wohlgemuth, W. K., Krystal, A. D., and Rice, J. R. (2005). Behavioral insom nia therapy for fi bromyalgia patients: a randomized clinical trial. Arch Int Med, 165, 252 7 \200\2232535 . Foley, D., Ancoli-Israel, S., Britz, P., and Walsh, J. (2004). Sleep disturbances and chron ic disease in older adults: results of the 2003 National Sleep Foundation Sleep in America Survey. J Psychosom Res , 56, 497 \200\223502 . Forest, G. and Godbout, R. (2000). Eff ects of sleep deprivation on performance and EEG spectral analysis in young adults. Brain Cogn , 43, 195 \200\223200 Forgeron, P. A., McGrath, P. J., Stevens, B., Evans, J., Dick, B. D., Finley, G. A., et al . (2011). Social information processing in adolescents with chronic p ain: my friends don \200\231t really understand me. Pain , 152(12), 2773 \200\2232780. Fric ke-Oerkermann, L., Pluck, J., Schredl, M., Heinz, K., Mitschke, A., and Wiater, A. (2007). Prevalence and course of sleep problems in childhood. Sleep , 30, 1371 \200\2231377 . Fuggle, P., Shand, P. A. X., Gill, L. J., and Davies, S. C. (1996). Pain, quality of life and coping in sickle cell disease. Arch Dis Child, 75, 199 \200\223203 . Gagliese, L. and Chambers, C. T. (2007). Pediatric and geriatric pain in relation to sleep disturbances. In G. Lavigne, B. J. Sessle, M. Choiniere and P. J. Soja (eds) Sleep and pain , pp. 341 \200\22359 . Seattle, WA : IASP Press . References Bloom, B. J., Owens, J. A., McGuinn, M., Nobile, C., Schaeff er, L., and Alario, A. J. (2002). Sleep and its relationship to pain, dysfunction, Gold, J. I., Mahrer, N. E., Yee, J., and Palermo, T. M. (2009). Pain, fatigue, and health-r elated quality of life in children and adolescents with chronic pain. Clin J Pain, 25 (5), 407 \200\223412 . SECTION 3 social and psychological basis of pediatric pain 142 Miller, V. A., Palermo, T. M., Powers, S. W., Scher, M. S., and Hershey, A. D. (2003). Migr aine headaches and sleep disturbances in children. Headache, 43, 362 \200\223368 . Henry, D. E., Chiodo, A. E., and Yang, W. (2011). Central nervous system reorganization in a variety of chronic pain states: a review. PM R, 12, 1116 \200\2231125 . Mindell, J. A., Emslie, G., Blumer, J., Genel, M., Glaze, D., Ivanenko, A., and Banas, B. ( 2006a). Pharmacologic management of insomnia in children and adolescents: Consensus stateme nt. Pediatrics, 117, e1223 \200\223e1232 . Mindell, J. A., Kuhn, B., Lewin, D. S., Meltzer, L. J., and Sadeh, A. (2006b). Behavioral treatment of bedtime problems and night wakings i n infants and young children. Sleep , 29, 1263 \200\2231276 . Jungquist, C. R., O \200\231Brien, C., Matteson-Rusby, S., Smith, M. T., Pigeon, W. R., Xia , Y., and Perlis, M. L. (2010). Th e effi cacy of cognitive-behavioral therapy for insomnia in patients with chronic pain. Sleep Med , 11 , 302 \200\223309 . Kashikar-Zuck, S., Flowers S. R., Claar, R. L., Guite, J. W., Logan, D. E., Lynch- Jordan, A.M., et al . (2011). Clinical utility and validity of the functional disability inventory among a multicenter sample of youth with chronic pain. Pain , 152, 1600 \200\2231607 . Mindell, J. A. and Owens, J. A. (2003). Sleep problems in pediatric practice: clinical issu es for the pediatric nurse practitioner. J Pediatr Health Care , 17(6), 324 \200\223331. Kashikar-Zuck, S., Flowers, S. R., Verkamp, E., Ting, T. V., Lynch-Jordan, A. M., Graham, T . B., et al . (2010). Actigraphy-based physical activity monitoring in adolescents with juv enile primary fi bromyalgia syndrome. J Pain , 11, 885 \200\22393 . Morin, C. M. (2010). Chronic insomnia: recent advances and innovations in treatment develop ments and dissemination. Can Psychol , 51, 31 \200\22339 . Owens, J. A. (2005). Epidemiolog y of sleep disorders during childhood. In S. H. Sheldon, R. Ferber, and M. H. Kryger (eds) Principles and practices of pediatric sleep medicine (4th edn), pp. 27 \200\22333 . Philade lphia, PA : Elsevier/ Saunders . Kashikar-Zuck, S., Johnston, M., Ting, T. V., Graham, B. T., Lynch-Jordan, A. M., Verkamp, E., et al . (2010). Relationship between school absenteeism and depressive symptoms among a dolescents with juvenile fi bromyalgia. J Pediatr Psychol, 35, 996 \200\2231004 . Owens J. A. (2007). Classifi cation and epidemiology of childhood sleep Kashikar-Zuck, S., Ting, T. V., Arnold, L. M., Bean, J., Powers, S. W., Graham, T. B., et a l . (2012). Cognitive behavioral therapy for the treatment of juvenile fi bromyalgia: a mul tisite, single-blind, randomized, controlled clinical trial. Arthritis Rheum, 64, 297 \200 \223305 . disorders. Sleep Med Clin , 2, 353 \200\223361 . Owens, J. A. (2011). Update in pediatric sleep medicine. Curr Opin Pulm Med, 17, 425 \200\223430 . Owens, J. A., Babcock, D., Blumer, J., Chervin, R., Ferber, R., Goetting, M., et al . (2005 ). Th e use of pharmacotherapy in the treatment of pediatric insomnia in primary care: rati onal approaches. A consensus meeting summary. J Clin Sleep Med , 15( 1), 49 \200\22359 . Kashikar-Zuck, S., Parkins, I. S., Ting, T. V., Verkamp, E., Lynch-Jordan, A., Passo, M., e t al . (2010). Controlled follow-up study of physical and psychosocial functioning of adole scents with juvenile primary fi bromyalgia syndrome. Rheumatology, 49, 2204 \200\2232209 . Owens, J. A. and Moturi, S. (2009). Pharmacologic treatment of pediatric Lewandowski, A. S., Palermo, T. M., De la Motte, S., and Fu, R. (2010). Temporal daily asso ciations between pain and sleep in adolescents with chronic pain versus healthy adolescents . Pain , 151(1), 220 \200\223225. insomnia. Child Adolesc Psychiatr Clin N Am, 18, 1001 \200\2231016 . Owens, J. A., Spirito, A., and McGuinn, M. (2000). Th e Children \200\231s Sleep Habits Que stionnaire (CSHQ): psychometric properties of a survey instrument for school-aged children. Sleep , 23(8), 1043 \200\2231051. Lewandowski, A. S., Toliver-Sokol, M., and Palermo, T. M. (2011a). Evidence- based review o f subjective pediatric sleep measures. J Pediatr Psychol , 36, 780 \200\223793 . Palermo, T. M. (2009). Assessment of chronic pain in children: current status and emerging topics. Pain Res Manag, 14, 21 \200\22326 . Lewandowski, A. S., Ward, T. M. and Palermo, T. M. (2011b). Sleep problems in children and adolescents with common medical conditions. Ped Clin N Am, 58, 699 \200\223713 . Palermo, T. M., Fonareva, I., and Janosy, N. R. (2008). Sleep quality and effi ciency in ad olescents with chronic pain: relationship with activity limitations and health-related qual ity of life. Behav Sleep Med, 6, 234 \200\223250 . Lewin, D. S. and Dahl, R. E. (1999). Importance of sleep in the management of pediatric pain. J Dev Behav Pediatr, 20 , 244 \200\223252 . Palermo, T. M. and Owens, J. (2008). Introduction to the special issue: sleep Long, A. C., Krishnamurthy, V., and Palermo, T. M. (2008). Sleep disturbances in school-age children with chronic pain. J Pediatr Psychol, 33, 258 \200\223268 . Long, A. C., Palermo, T. M., and Manees, A. M. (2008). Brief report: using actigraphy to compare physical activi ty levels in adolescents with chronic pain and healthy adolescents. J Pediatr Psychol, 33, 660 \200\223665 . in pediatric medical populations. J Pediatr Psychol, 33, 227 \200\223231 . Palermo, T. M., Toliver-Sokol, M., Fonareva, I., and Koh, J. L. (2007). Objective and subje ctive assessment of sleep in adolescents with chronic pain compared to healthy adolescents. Clin J Pain, 23, 812 \200\223820 . Palermo, T. M., Wilson, A. C., Lewandowski, A. S., Toliver-Sokol, M., and Murray, C. B. (20 11). Behavioral and psychosocial factors associated with insomnia in adolescents with chron ic pain. Pain, 152, 89 \200\22394 . Palermo, T. M., Wilson, A. C., Peters, M., Lewandowski, A., and Somhegyi, H. (2009). Randomized controlled trial of an internet-delivered family c ognitive \200\223behavioral therapy intervention for children and adolescents with chronic pain. Pain , 146, 205 \200\223213 . Maynard, C. S., Amari, A., Wieczorek, B., Christensen, J. R., and Slifer, K. J. (2010). Int erdisciplinary behavioral rehabilitation of pediatric pain- associated disability: retrospe ctive review of an inpatient treatment protocol. J Ped Psych , 35, 128 \200\223137 . Martin, J., Hiscock, H., Hardy, P., Davey, B., and Wake, M. (2007). Adverse associations of infant and child sleep problems and parent health: an Australian population study. Pediatr ics, 119 (5), 946 \200\223955 . Pirinen, T., Kolho, K. L., Simola, P., Ashorn, M., and Aronen, E. T. (2010). Parent and sel f-report of sleep-problems and daytime tiredness among adolescents with infl ammatory bowel disease and their population- based controls. Sleep , 33(11), 1487 \200\2231493. Meltzer, L. J., Johnson, C., Crosette, J., Ramos, M., and Mindell, J. A. (2010). Prevalence of diagnosed sleep disorders in pediatric primary care practices. Pediatrics , 125 , e1410 \200\223e1418 . Meltzer, L. J., Logan, D. E., and Mindell, J. A. (2005). Sleep patterns in female adolescen ts with chronic musculoskeletal pain. Behav Sleep Med, 3, 193 \200\223208 . Rybarczyk, B., Stepanski, E., Fogg, L., Lopez, M., Barry, P., and Davis, A. (2005). A place bo controlled test of cognitive-behavioral therapy for comorbid insomnia in older adults. J Consult Clin Psychol, 73, 1164 \200\2231174 . Meltzer, L. J. and Mindell, J. A. (2009). Pediatric sleep. In M. C. Roberts and R. G. Steel e (eds) Handbook of pediatric psychology (4th edn), pp. 491 \200\223 507 . New York : Th e Guilford Press . Sadeh, A. (2007). Consequences of sleep loss or sleep disruption in children. Sleep Med Clinics , 2, 513 \200\223520 . Saper, C. B., Cano, G., and Scammell, T. E. (2005). Homeostatic, circadian, Meltzer, L. J. and Westin, A. M. (2011). A comparison of actigraphy scoring and emotional regulation of sleep. J Comp Neurol, 493, 92 \200\22398 . rules used in pediatric research. Sleep Med, 12, 793 \200\223796 . Sivertsen, B., Hysing, M., Elgen, I., Stormark, K., and Lundervold, A. (2009). Chronicity o f sleep problems in children with chronic illness: a longitudinal population-based study. C hild Adol Psychiat Ment Health, 3, 1 \200\2237 . Menefee, L. A., Cohen, M. M., Anderson, W. R., Doghramji, K., Frank, E. D., and Lee, H. (20 00). Sleep disturbance and nonmalignant chronic pain: a comprehensive review of the literat ure. Pain Med , 1, 156 \200\223172 . CHAPTER 15 sleep and pain in children and adolescents 143 Vendrame, M., Kaleyias, J., Valencia, I., Legido, A., and Kothare, S.V. (2008). Polysomnogr aphic fi ndings in children with headaches. Pediatr Neurol, 39 , 6 \200\22311 . Smith, M. T., Huang, M. I., and Manber, R. (2005). Cognitive behavior therapy for chronic i nsomnia occurring within the context of medical and psychiatric disorders. Clin Psychol Rev , 25, 559 \200\223592 . Vendrame, M., Zarowski, M., Loddenkemper, T., Steinborn, B., and Kothare, S.V. (2011). Sele ctive serotonin reuptake inhibitors and periodic limb movements of sleep. Pediatr Neurol, 4 5, 175 \200\223177 . Stiefel, F. and Stagno, D. (2004). Management of insomnia in patients with chronic pain conditions. CNS Drugs , 18, 285 \200\223296 . Tayag-Kier, C. E., Keenan, G. F., Scalzi, L. V., Schultz, B., Elliott, J., Zhao, R. H., et al . (2000). Sleep and periodic limb movement in sleep in juvenile fi bromyalgia. Pediatric s, 106, E70 . Vriend, J. and Corkum, P. (2011). Clinical management of behavioral insomnia of childhood. Psych Res Beh Man, 4, 69 \200\22379 . Vriend, J. L., Davidson, F. D., Corkum, P. V., Rusak, B., McLaughlin, E. N., and Chambers, C. T. (2012). Sleep quantity and quality in relation to daytime functioning in children. Ch ild Health Care, 41(3), 204 \200\223222. Ward, T. M., Brandt, P., Archbold, K., Lentz, M., Ringold, S., Wallace, C. A., et al . (2008). Polysomnography and self-reported sleep, pain, fatigue, and anxiety in children with active and inactive juvenile rheumatoid arthritis. J Pediatr Psychol, 33, 232 \200\223241 . Tsai, S. Y., Labyak, S. E., Richardson, L. P., Lentz, M. J., Brandt, P. A., Ward, T. M., et al . (2008). Actigraphic sleep and daytime naps in adolescent girls with chronic musculosk eletal pain. J Pediatr Psychol, 33, 307 \200\223311 . Valrie, C. R., Gil, K. M., Redding-La llinger, R., and Daeschner, C. (2008). Brief report: daily mood as a mediator or moderator of the pain-sleep relationship in children with sickle cell disease. J Pediatr Psychol, 33, 317 \200\223322 . Witmans, M. B., Dick, B., Good, J., Schoepp, G., Dosman, C., Hawkins, M., and Witol, A. (20 08). Delivery of pediatric sleep services via telehealth: the Alberta experience and lesson s learned. Behav Sleep Med , 6, 207 \200\223219 . Valrie, C. R., Gil, K. M., Redding-Lallinger, R., and Daeschner, C. (2007). Th e infl uence of pain and stress on sleep in children with sickle cell disease. Child Health Care, 36, 3 35 \200\22353 . This page intentionally left blank SECTION 4 Pain in speci \201 c populations and diseases This page intentionally left blank CHAPTER 16 Pain in children with intellectual or developmental disabilities John L. Belew , Chantel C. Barney, Scott A. Schwantes, Dick Tibboel, Abraham J. Valkenburg , and Frank J. Symons Summary In this chapter, it is argued that it is time to bring the burgeoning work on pain together and develop a coherent clinically-informed research agenda addressing the problem of pain among children with intellectual or developmental disabilities (I/DD). Th e chapter starts with an introductory section providing a short overview of the problem of pain amon g children with I/DD including many defi nitional and historical issues. Subsequent section s include a review of the epidemiology and impact of pain in children with I/DD, followed b y a review of pain assessment and then pain man- agement in children with I/DD. Th e chapte r ends with a summary and conclusions relevant for future research and practice. Pain \200\224important and prevalent in children with I/DD Th ere is little doubt that pain is a universal phenomenon causing tremendous human suff ering and compromising the quality of life for countless individuals. Th e US congress declared 2001 to 2010 as the Decade of Pain Control and Research, however we estimate a decade devoted to pain research yielded f ewer than 80 papers about pain in children with I/DD. And yet, there is no good reason to b elieve that pain is any less frequent in a child with a developmen- tal disability or intel lectual impairment, or that such an individual would be insensitive or indiff erent to pain . Numerous functional limitations as well as the underlying neurological condition itself f requently confound the presentation of pain. Regardless of the degree of the disability, ho wever, pain is oft en a part of daily life for children with I/DD (Oberlander and Symons, 2 006). Introduction Th ere is an enormous amount of scientifi c data about pain but com- parativel y little knowledge specifi c to children with intellectual or developmental disability. A P ubMed search using the terms \200\230pain \200\231 and \200\230human \200\231 yielded ove r 100 000 publications for the 5-year period between 2005 and 2009 (the last full 5-year ep och) or, approximately 20 000 papers per year reporting on new discoveries, reviews, and th eories regarding pain and the human condition. Only a fraction (3.7%) of those papers addre ssed pediatrics and pain (the topic of this volume). For that same 5-year period, there wer e a total of 30 scientifi c papers published specifi c to pain and children with I/DD \200 \224approximately 0.03% of the publication volume. Considering that approximately 1% to 7% of the world \200\231s population lives with an intellectual or related developmental disab ility (estimates vary widely between developed and developing countries; World Health Organ ization, 2001), the gap between what science has to tell us about pain in this population a nd what we probably need to know is striking (Figure 16.1 ). A contributing factor for this paucity of literature is most likely the routine exclusion of individuals with I/DD from r esearch. Th e majority of scientifi c activity devoted to pain and children with I/DD in the previou s decade has been to \200\230build a better mousetrap \200\231 \200\224 improving behavior al assessment techniques to identify accurately the presence of pain and its source or type , when reliable self-report is not an option. Th e fruits of this labor are manifest in sev eral rating scales and questionnaires designed to measure pain intensity/sever- ity and its expression (these are reviewed in detail in later sections). Although the terms intellectu al disability and developmental dis- ability are familiar ones, we encourage the reader to pay careful attention to the explanation of these terms. Th e term I/DD includes a populati on of children that is highly heterogeneous: children with conditions of diverse type and e tiology (e.g. Down syndrome, cer- ebral palsy (CP), Rett syndrome) are included along with children with neurodevelopmental disabilities of unknown origin. Th erefore our attempt her e is to portray common features related to pain expe- rience and management that are found across this diverse group, and specifi cally within certain subpopulations. By the very nat ure of their underlying condition children with I/DD are particularly at risk for painful e xperiences disproportionate to their typically developing peers. Common vulnerabilities chi ldren with I/DD face SECTION 4 pain in specific populations and diseases 148 in intellectual functioning and in adaptive behavior, which covers many everyday social and practical skills. Th is disability originates before the age of 18 \200\231 (2012). Simila rly, the International Association for the Scientifi c Study of Intellectual Disability (IA SSID) considers ID as a \200\230signifi cant intellectual defi cit present from birth, or which originates at an early age or during the developmental period \200\231 (2012). Th ere are key components to the defi nitions specifi c to the concept of \200\230intellectual f unctioning \200\231 and \200\230adaptive behavior \200\231. Intellectual functioning (i.e. intelligence) \200\230refers to general mental capacity, such as learning, reasoning, pro blem solving, and so on. One criterion to measure intellectual functioning is an IQ test. G enerally, an IQ test score of around 70 or as high as 75 indicates a limitation in intel- l ectual functioning \200\231 [i.e. greater than two standard deviations below average IQ] (A AIDD, <http://www.aaidd.org/content_100.cfm>). A similar approach is used to determine adap tive function. Specifi cally, standardized tests are used to determine limitations in adapt ive behavior, comprised of three specifi c skill types includ- ing conceptual skills (e.g. number concepts), social skills (e.g. social problem solving), and practical skills (e.g. a ctivities of daily living). On the basis of a set of standardized measures designed to spec ifi - cally assess and evaluate intelligence and adaptive behavior it is determined whether a child has an ID (see Salvador-Carrulla et al. 2011 for an interesting discussion of ID i n the medical context.) (a) 90,000 80,000 70,000 60,000 50,000 40,000 30,000 20,000 10,000 0 Pediatric Pain Pediatric Pain \200\230Plus \200\231 Pain Human 1960 \200\22364 1975 \200\22379 1965 \200\22369 1970 \200\22374 1980 \200\22384 1985 \200\22389 1990 \200\22394 1995 \200\22399 2000 \200\22304 2005 \200\22309 (b) 4,500 4,000 3,500 3,000 2,500 2,000 1,500 1,000 500 0 Pediatric Pain Pediatric Pain \200\230Plus \200\231 De \201 ning developmental disability Developmental disability as defi ned in the US by th e Developmental Disabilities Assistance and Bill of Rights Act (DD Act; PL. 98 \200\223527) refers to a \200\230severe, chronic disability \200\231 attributable to \200\230mental \200\231 and/or \200\230physi- cal \200\231 impairments that are \200\230likely to contin ue indefi nitely \200\231, resulting in substantial functional imitations in three or more \200\230major life activity areas: self-care, receptive or expressive language, learning, self-direc- tion, capacity for independent living and economic self-suffi ciency \200\231, are manifest by age 22 and require care, treatment or other services of lifelong extended d uration. Some conditions may be included cat- egorically, e.g. CP, spina bifi da, autism. I t is worth noting that unlike \200\230intellectual disability \200\231, classifying develo pmental disability does not benefi t from a prescribed set of standardized assessment instr uments, procedures, or professional training programs. From a somewhat similar perspective, through the World Health Organization (WHO), Th e International Classifi cation of Functio ning, Disability and Health (referred to as ICF) also provides a classifi cation system for health and health-related domains aff ected by disability. Th e domains are clas- sifi ed from body, individual and societal perspectives in two areas; (1) body functions and struct ure, and (2) activity and participation. Th e ICF is WHO \200\231s framework for measuring health and disability at both individual and population levels. 1960 \200\22364 1965 \200\22369 1970 \200\22374 1980 \200\22384 1985 \200\22389 1990 \200\22394 1995 \200\22399 2000 \200\22304 2005 \200\22309 1975 \200\22379 Figure 16.1 Cumulative frequency of pediatric pain research based on PubMed search results for full 5-year epochs using search terms \200\230pain \200\231, \200\230human \200\231, \200\230pediatric \200\231, and \200\230pediatric \200\231 plus \200\230intellectual dis ability \200\231, \200\230mental retardation \200\231, \200\230developmental disability ( a) and results for \200\230pain \200\231, \200\230pediatric \200\231, and \200\230pediat ric plus \200\231 only (b). with respect to pain include increased prevalence of painful condi- tions, both acute and c hronic, higher rate of exposure to painful procedures (diagnostic and therapeutic), and neu rosensory con- cerns that heighten the pain experience as well as complicate pain assessmen t and management.