527501
research-article2014
CPJXXX10.1177/0009922814527501Clinical PediatricsBodenner et al
Article
Assessment and Treatment of Obstructive Sleep-Disordered Breathing
Clinical Pediatrics 2014, Vol. 53(6) 544–548 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0009922814527501 cpj.sagepub.com
Kaitlyn Anne Bodenner1, Supriya K. Jambhekar, MD2, Gulnur Com, MD2, and Wendy L. Ward, PhD2,3
Abstract Obstructive sleep-disordered breathing (OSDB) is a condition that affects 1% to 3% of the pediatric population. These disorders are difficult to diagnosis and left untreated may be serious, including not only medical comorbidities but also cognitive, academic, behavioral, and emotional sequelae. This article is designed to bring awareness of the severity and prevalence to family physicians and pediatricians. It reviews detailed information concerning OSDB, including the predisposing factors, assessment of presenting features, and treatment. Keywords obstructive sleep apnea, sleep-disordered breathing, pediatric sleep disorder
Introduction Obstructive sleep-disordered breathing (OSDB) is defined as obstruction of the upper airway during sleep that negatively affects sleep quality.1 “This may involve either complete upper airway obstruction (apnea), repeated episodes of partial upper airway obstruction (hypopnea), or prolonged increased upper airway resistance syndrome (UARS).”1(p65) OSDB is thought to be caused by “either anatomic narrowing of the upper airway, or increased collapsibility of UAW muscles, or abnormal neural control; usually a combination of all three events.”1(p67) Recently, the American Academy of Pediatrics2 released guidelines on the detection of OSDB in primary care settings, given findings that symptoms of OSDB may be missed, which can result in many adverse outcomes if left untreated. In addition, it has been shown that there is no correlation between symptoms and clinic findings and OSDB.3 Obstructive events during sleep are not easily identified in the general pediatrics setting and may result in many adverse outcomes if left untreated.4 In fact, given the range and severity of comorbidities associated with OSDB, it is essential that pediatric practitioners be aware of this condition and screen all children at well-child visits for the condition.5 Family physicians and pediatricians need to be aware of the disorder in children, the presenting features, the comorbidities, the predisposing factors, and the need for screening and referral
Why Obstructive Sleep-Disordered Breathing Is a Problem Obstructive sleep-disordered breathing affects approximately 1% to 3% of the pediatric population.6 With an estimated pediatric population of 74.2 million,7 there could be an estimated 1.5 to 2.2 million with OSDB in the United States. If untreated, OSDB results in a variety of negative outcomes. Medical complications include, impaired somatic growth, nocturnal enuresis, failure to thrive, and possible neuronal injury in the hippocampus and frontal cortex.8-11 OSDB in children has been linked to impaired cardiac function and structure,12 and other cardiovascular complications including impaired right ventricular function and cor pulmonale.13,14 Furthermore, OSDB has been associated with blood pressure dysregulation in some but not all studies.15 Circulating inflammatory mediators and increased insulin resistance have been posited as one of the potential process mechanisms, especially in obese children.16,17 OSDB may also be associated with extraesophageal reflux,18 lower exhaled
1
University of Central Arkansas, Conway, AR, USA University of Arkansas for Medical Sciences, Little Rock, AR, USA 3 Arkansas Children’s Hospital, Little Rock, AR, USA 2
Corresponding Author: Wendy L. Ward, Arkansas Children’s Hospital, 1 Children’s Way, Slot 512-21, Little Rock, AR 72202, USA.
545
Bodenner et al
Table 1. Questions Physicians Should Ask Patients or Caregivers for Assessment of Obstructive Sleep-Disordered Breathing.a Questions About Nighttime Manifestations Does he or she snore? Does he or she have uneven breathing? Does he or she have trouble breathing while sleeping? Have you ever witnessed them choke or make loud gasping sounds during the night? Is it difficult to awake your child? Does your child often wet the bed? How would you describe their sleeping position? (strange sleeping position) Does he or she sweat profusely while they are asleep? Is their sleep restless or do they startle awake?
Questions About Daytime Manifestations Do they awake with morning headaches or vomiting? Is he or she doing well in school? If not, has there been a recent onset of school complaints? Does he or she have attention problems or difficulty focusing? Has your child exhibited a depressed mood? Does the child have low appetite on wakening? Does he or she have memory problems? Have you noticed hyperactivity, irritable mood, or any other behavioral problems? Does he or she seem excessively tired during the daytime?
a
This table was adapted from one published in 2010.
nitric oxide/less eosinophilic and more neutrophilic type of asthma,19 and acutely decompensated heart failure.20 A causal relationship between nighttime sleep fragmentation and daytime neurobehavioral dysfunction has been suggested by multiple studies.9,21-24 Neurobehavioral dysfunctions associated with OSDB include poor school performance, cognitive dysfunction, inattentiveness, impaired memory, IQ deficit, executive function abnormalities, and so on.10,25-27 Behavioral outcomes are common as well—hyperactivity, conduct disorder, aggressive behavior, and so forth.25,26 Emotional outcomes are also present, for instance, quality-of-life impairment and depressive symptoms.28 Recent studies have also linked the neurobehavioral consequences of OSDB in children to inflammatory markers. However, these relationships are complex, as some circulating factors are associated with poor outcomes and others are protective.26,29,30
Predisposing Factors Enlarged tonsils and adenoids are the most common cause of OSDB in children.31-35 Obesity is another risk factor for OSDB.36,37 The reported prevalence of OSDB in obese youth ranges from 37% to 66%.38-40 Other conditions increasing the likelihood of OSDB are genetic syndromes, including Down syndrome and Prader–Willi syndrome; craniofacial anomalies, including syndromes such as Apert syndrome or Crouzon syndrome, achondroplasia, Arnold–Chiari malformation; neuromuscular syndromes, including muscular dystrophy; and cerebral palsy due to muscle tone abnormalities.
Assessment and Presenting Features Patients present a wide variety of nonspecific symptoms that make identification of the disorder challenging. The
American Academy of Pediatrics has presented guidelines on assessment of obstructive sleep apnea syndrome, a condition involving OSDB and impaired daytime functioning.41 This document suggests that pediatric patients may present with daytime manifestations of OSDB, including the cognitive, school, emotional, and behavioral comorbidities noted above. The symptoms include snoring, whistling noises, uneven/ jagged breathing while sleeping, occasional choking, and/or unusual sleep positions (often with the neck hyperextended). In addition, patients present with a myriad of sleep concerns—startling awake, restless sleep, profuse sweating. In the morning, they may have difficulty awakening or irritability on waking, morning headaches, vomiting, and lack of appetite for breakfast due to hypoventilation. Diagnosing OSDB first requires taking a detailed medical history, including daytime symptoms, nighttime symptoms, and adverse outcomes as described above (see Table 1) followed by physical examination. Radiological evaluation, including neck X-ray to look for enlarged adenoids, may help with diagnosis. Magnetic resonance imaging of the brain may be ordered in some patients to rule out foramen magnum stenosis or Arnold–Chiari malformation. X-ray cephalometry, computed tomography scanning and magnetic resonance imaging, dynamic scanning protocols (ultrafast computed tomography or cine magnetic resonance imaging), upper airway endoscopy during sleep, pressure measurements for the critical closing pressure are some other methods that may be used in selected patients.42-45 Further referral to a pediatric sleep center may be necessary for comprehensive assessment. Diagnosis is greatly supported by overnight polysomnography (PSG). PSGs assess “airflow at nose and mouth, thoracic and abdominal wall movements to detect obstructive respiratory events, oximetry for oxygenation, and end tidal CO2
546 to assess ventilation.”1(p6) Electroencephalography and electro-oculogram channels monitor stages of sleep throughout the study. Other conditions like periodic limb movements during sleep, nocturnal seizures, and parasomnias can also be detected on PSG. However, the PSG alone cannot be the sole assessment tool for several reasons. First, there are no proven norms for children and the exact significance of abnormal numbers in terms of predictive value for adverse outcome or need for treatment is not clearly defined. Second, significant neurobehavioral outcomes have been linked to clinical symptoms of OSDB without classical findings on PSG.46 Hence, diagnosis of OSDB and decision to treat are made based on a combination of all approaches.
Treatment of Obstructive Sleep-Disordered Breathing Once the diagnosis of OSDB is made and a decision to treat is taken, nasal steroids or leukotriene antagonists are helpful especially with upper airway congestion and mild adenoidal growth.47,48 It should be noted that while melatonin, antidepressants, and nonbenzodiazepine sleep-inducing medications may be used to help the patient sleep, they cannot be used as a sleep aid until the disordered breathing during sleep is improved and may in fact exacerbate OSDB. Tonsillo-adenoidectomy is usually the first line of treatment for children who have enlarged tonsils and adenoids and has been shown to help in a majority of these children.49 A wide range of additional surgical options (uvulopalatopharyngoplasty, maxillomandibular advancement, etc) are available as appropriate. However, a significant percentage of children continue to have OSDB following surgery50,51 or are poor candidates for surgery. This population includes mainly obese children49 and youth with craniofacial anomalies or neuromuscular difficulties.1 These children require treatment with positive airway pressure (PAP), either continuous positive airway pressure (CPAP) or bilevel positive airway pressure (B-PAP). CPAP has been shown to be effective in controlling the respiratory events and improving sleep parameters in children.53-58 “The main limitation of PAP use is poor adherence.”1(p68) Adherence studies in adults have shown that 30% to 46% patients with OSDB discontinue the use of PAP against medical advice.59,60 Emerging evidence regarding pediatric compliance with PAP treatment is available.53,54 It has been thought that early intensive intervention, including behavioral techniques such as systematic desensitization and reinforcement strategies will help in improving adherence with PAP use.53,59,61-63 Some patients describe difficulty initially tolerating their mask due to a claustrophobic reaction to the mask, significant sensory sensitivities, which
Clinical Pediatrics 53(6) may decrease tolerance to the feel of the equipment or the air pressure, or general unfamiliarity with the equipment, which may induce fear reactions. Children with developmental delays also have lowered adherence rates on average, as do individuals with disruptive behaviors.64 Structured adherence programs for PAP use are available and effective.65 Referral to a pediatric sleep center may be necessary for comprehensive management in some children. Oral appliances that attempt to increase upper airway size are used successfully in adults44,66; however, there is very little experience in children.67 The last option is tracheostomy, which is often effective but is considered highly invasive. In addition, tracheostomy is associated with increased morbidity and mortality.68
Conclusion In summary, OSDB is a common and often undetected condition that has a wide-ranging impact on patient functioning, including not only medical comorbidities but also cognitive, academic, behavioral, and emotional sequelae. Family physicians and pediatricians need training and education on sleep disorders, crucial in identification and treatment of pediatric patients with OSDB, to prevent these myriad health effects. Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
References 1. Jambhekar SK, Com G, Ward-Bengnoche W. Obstructive sleep disordered breathing in children: assessment and treatment at Arkansas Children’s Hospital. J Ark Med Soc. 2010;107:65-68. 2. Marcus CL, Brooks LJ, Draper KA, et al. American Academy of Pediatrics. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2012;130:576-584. 3. Carroll JL, McColley SA, Marcus CL, Curtis S, Loughlin GM. Inability of clinical history to distinguish primary snoring from obstructive sleep apnea syndrome in children. Chest. 1995;108:610-618. 4. Park JG, Ramar K, Olson EJ. Updates on definition, consequences, and management of obstructive sleep apnea. Mayo Clin Proc. 2011;88:549-555. 5. Brown JG, Schnall P, Hallgren JP. Clinical inquires. When (and how) should you evaluate a child for obstructive sleep apnea? J Fam Pract. 2007;56:317-320.
Bodenner et al 6. Faruqui F, Khubchandani J, Price JH, Bolyard D, Reddy R. Sleep disorders in children: a national assessment of primary care pediatrician practices and perceptions. Pediatrics. 2011;128:539-546. 7. Howden L, Meyer J. US Department of Commerce, Economics and Statistics Administration. Age and Sex Composition: 2010 (C2010BR-03). Washington, DC: US Census Bureau; 2011. 8. Chhangani BS, Melgar T, Patel D. Pediatric obstructive sleep apnea. Indian J Pediatr. 2009;77:81-85. 9. Beebe DW, Gozal D. Obstructive sleep apnea and the prefrontal cortex: towards a comprehensive model linking nocturnal upper airway obstruction to daytime cognitive and behavioral deficits. J Sleep Res. 2002;11:1-16. 10. Halbower AC, Degaonkar M, Barker PB, et al. Childhood obstructive sleep apnea associates with neuropsychological deficits and neuronal brain injury. PLoS Med. 2006;3(8):e301. 11. Section on Pediatric Pulmonology, Subcommittee on Obstructive Sleep Apnea Syndrome. American Academy of Pediatrics. Clinical practice guideline: diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2002;109:704-712. 12. Amin R, Carroll JL, Jeffries JL, et al. Twenty-four hour ambulatory blood pressure in children with sleepdisordered breathing. Am J Respir Crit Care Med. 2004;169:950-956. 13. Amin RS, Kimball TR, Bean JA, et al. Left ventricular hypertrophy and abnormal ventricular geometry in children and adolescents with obstructive sleep apnea. Am J Respir Crit Care Med. 2002;165:1395-1399. 14. Gozal D, Kheirandish-Gozal L. Neurocognitive and behavioral morbidity in children with sleep disorders. Curr Opin Pulm Med. 2007;13:505-509. 15. Zintzaras E, Kaditis AG. Sleep-disordered breathing and blood pressure in children: a meta-analysis. Arch Pediatr Adolesc Med. 2007;161:172-178. 16. Flint J, Kothare SV, Zihlif M, et al. Association between inadequate sleep and insulin resistance in obese children. J Pediatr. 2007;150:364-369. 17. Gozal D, Serpero LD, Capdevila OS, Kheirandish-Gozal L. Systemic inflammation in non-obese children with obstructive sleep apnea. Sleep Med. 2008;9:254-259. 18. Demeter P, Pap A. The relationship between gastro esophagel reflux disease and obstructive sleep apnea. J Gasteoenterol. 2004;39:815-820. 19. Kheirandish-Gozal L. Asthma and obstructive sleep apnea. In:Kheirandish-Gozal L, Gozal D, eds. Sleep Disordered Breathing in Children: A Comprehensive Clinical Guide to Evaluation and Treatment. New York, NY: Humana Press/Springer; 2012:385-390. 20. Khayat R, Jarjoura D, Patt B, Yamokoski T, Abraham W. In-hospital testing for sleep disordered breathing in hospitalized patients with decompensated heart failure—report of prevalence and patient characteristics. J Card Fail. 2009;15:739-746. 21. Beebe DW. Neurobehavioral morbidity associated with disordered breathing during sleep in children: a comprehensive review. Sleep. 2006;29:1115-1134.
547 22. Sedky R, Carvalho KS, Lippmann S. Attention deficit hyperactivity disorder and sleep disordered breathing in children. J Pediatr Biochem. 2013;3:61-67. 23. Kheirandish L, Gozal D. Neurocognitive dysfunction in children with sleep disorders. Dev Sci. 2006;9:388-399. 24. Moreau V, Rouleau N, Morin CM. Sleep of children with attention deficit hyperactivity disorder: actigraphic and parental reports. Behav Sleep Med. 2014;12:69-83. 25. Beebe DW, Ris MD, Kramer ME, Long E, Amin R. The association between sleep disordered breathing, academic grades, and cognitive and behavioral functioning among overweight subjects during middle to late childhood. Sleep. 2010;33:1447-1456. 26. Gozal D, Crabtree VM, Capdevilla OS, Witcher LA, Kheirandish-Gozal L. C-reactive protein, obstructive sleep apnea, and cognitive dysfunction in school-aged children. Am J Respir Crit Care Med. 2007;176:188-193. 27. Schechter MS. Subcommittee on Obstructive Sleep Apnea Syndrome, Pediatric Pulmonology. Technical report: diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2002;109:e69. 28. Crabtree V, Varni J W, Gozal D. Health-related quality of life and depressive symptoms in children with suspected sleep-disordered breathing. Sleep. 2004;27:1131-1138. 29. Gozal D, Sans Capdevila O, McLaughlin Crabtree V, Serpero LD, Witcher LA, Kheirandish-Gozal L. Plasma IGF-1 levels and cognitive dysfunction in children with obstructive sleep apnea. Sleep Med. 2009;10:167-173. 30. Li AM, Chan MH, Yin J, et al. C-reactive protein in children with obstructive sleep apnea and the effects of treatment. Pediatr Pulmonol. 2008;43:34-40. 31. Brietzke SE, Kezirian E, Thaler ER, Woodson BT. Novel sleep apnea surgical treatments. Otolaryngol Head Neck Surg. 2012;147(2 suppl):P34. 32. Marcus CL, Carole L. Sleep-disordered breathing in children. Am J Respir Crit Care Med. 2001;164:16-30. 33. Marcus CL. Obstructive sleep apnea syndrome: differences between children and adults. Sleep. 2000;23(suppl 4):S140-S141. 34. Marcus CL, Carole L. Sleep-disordered breathing in children. Curr Opin Pediatr. 2000;12:208-212. 35. Katz N, Tamar E, Pillar G. Functional aspects and upper airway control during wakefulness and sleep. In:Kheirandish-Gozal L, Gozal D, eds. Sleep Disordered Breathing in Children: A Comprehensive Clinical Guide to Evaluation and Treatment. New York, NY: Humana Press/Springer; 2012:13-24. 36. Maalej S, Aouadi S, Ben Moussa H, Bourguiba M, Ben Kheder A, Drira I. Predictive factors of obstructive sleep apnea syndrome in obesity [in French]. Tunis Med. 2010;88:92-96. 37. O’Gorman S, Horlocker T, Huddleston J, Gay P, Morgenthaler T. Does self-titrating CPAP therapy improve postoperative outcome in patients at risk for obstructive sleep apnea syndrome? A randomized controlled clinical trial. Chest. 2011;140(4_Meeting Abstracts):1071A. 38. Lam YY, Chan EY, NG DK, et al. The correlation among obesity, apnea-hypopnea index, and tonsil size in children. Chest. 2006;130:1751-1756.
548 39. Marcus CL, Curtis S, Koerner CB, Joffe A, Serwint JR, Loughlin GM. Evaluation of pulmonary function and polysomnography in obese children and adolescents. Pediatr Pulmonol. 1996;21:176-183. 40. Silvestri JM, Weese-Mayer DE, Bass MT, Kenny AS, Hauptman SA, Pearsall SM. Polysomnography in obese children with a history of sleep-associated breathing disorders. Pediatr Pulmonol. 1993;16:124-129. 41. Marcus CL, Brooks LJ, Draper KA, et al; American Academy of Pediatrics. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2012;130:e714-e755. 42. Maurer JT, Stuck BA. Update on upper airway evaluation in obstructive sleep apnea [in German]. HNO. 2008;56:1089-1097. 43. Stuck BA, Maurer JT. Airway evaluation in obstructive sleep apnea. Sleep Med Rev. 2008;12:411-436. 44. De Backer JW, Vos WG, Verhulst SL, De Backer W. Novel imaging techniques using computer methods for the evaluation of the upper airway in patients with sleepdisordered breathing: a comprehensive review. Sleep Med Rev. 2008;12:437-447. 45. Abramson Z, Susaria S, Troulis M, Kaban L. Age-related changes of the upper airway assessed by 3-dimensional computed tomography. J Craniofac Surg. 2009;20 (suppl 1):657-663. 46. Gottlieb DJ, Chase C, Vezina RM, et al. Sleep-disordered breathing symptoms are associated with poorer cognitive function in 5-year-old children. J Pediatr. 2004;145:458-464. 47. Kheirandish L, Goldbart AD, Gozal D. Intranasal steroids and oral leukotriene modifier therapy in residual sleepdisordered breathing after tonsillectomy and adenoidectomy in children. Pediatrics. 2006;117:e61-e66. 48. Goldbart AD, Goldman JL, Veling MC, Gozal D. Leukotriene modifier therapy for mild sleep-disordered breathing in children. Am J Respir Crit Care Med. 2005;172:364-370. 49. Mitchell RB, Kelly J. Outcome of adenotonsillectomy for obstructive sleep apnea in obese and normal-weight children. Otolaryngol Head Neck Surg. 2007;137:43-48. 50. Guilleminault C, Huang YS, Glamann C, Li K, Chan A. Adenotonsillectomy and obstructive sleep apnea in children: a prospective survey. Otolaryngol Head Neck Surg. 2007;136:169-175. 51. Mitchell RB. Adenotonsillectomy for obstructive sleep apnea in children: outcome evaluated by pre- and postoperative polysomnography. Laryngoscope. 2007;117:1844-1854. 52. Downey R, Perkin RM, MacQuarrie J. Nasal continuous positive airway pressure use in children with obstructive sleep apnea younger than 2 years of age. Chest. 2000;117:1608-1612. 53. Marcus CL, Rosen G, Davidson-Ward SL, et al. Adherence to and effectiveness of positive airway pressure therapy in
Clinical Pediatrics 53(6) children with obstructive sleep apnea. Pediatrics. 2006;117: e442-e451. 54. Massa F, Gonsalez S, Laverty A, Wallis C, Lane R. The use of nasal continuous positive airway pressure to treat obstructive sleep apnea. Arch Dis Child. 2002;87: 438-443. 55. Popatia R, Rosen D. Infant obstructive sleep apnea successfully treated with continuous positive airway pressure [published online March 4, 2013]. Clin Pediatr (Phila). doi:10.1177/0009922813479166. 56. Palombini L, Pelayo R, Guilleminault C. Efficacy of automated continuous positive airway pressure in children with sleep-related breathing disorders in an attended setting. Pediatrics. 2004;113:e412-e417. 57. Waters KA, Everett F, Bruderer J, MacNamara F, Sullivan CE. The use of nasal CPAP in children. Pediatr Pulmonol. 1995;19(suppl 11):91-93. 58. Waters KA, Everett FM, Bruderer JW, Sullivan CE. Obstructive sleep apnea: the use of nasal CPAP in 80 children. Am J Respir Crit Care Med. 1995;152:780-785. 59. Edinger JD, Radtke RA. Use of in vivo desensitization to treat a patient’s claustrophobic response to nasal CPAP. Sleep. 1993;16:678-680. 60. Wolkove N, Baltzan M, Kamel H, Darbrusin R, Palayew M. Long-term compliance with continuous positive airway pressure in patients with obstructive sleep apnea. Can Respir J. 2008;15:365-369. 61. Kirk VG, O’Donnell AR. Continuous positive airway pressure for children: a discussion on how to maximize compliance. Sleep Med Rev. 2006;10:119-127. 62. Koontz KL, Slifer KJ, Cataldo MD, Marcus CL. Improving pediatric compliance with positive airway pressure therapy: the impact of behavioral intervention. Sleep. 2003;26:1010-1015. 63. Rains JC. Treatment of obstructive sleep apnea in pediatric patients. Behavioral intervention for compliance with nasal continuous positive airway pressure. Clin Pediatr (Phila). 1995;34:535-541. 64. Harford KL, Jambhekar S, Com G, et al. Behaviorally based adherence program for pediatric patients treated with positive airway pressure. J Clin Child Psychol Psychiatry. 2012;18:151-163. 65. Harford KL, Jambhekar S, Com G, et al. An in-patient model for positive airway pressure desensitization: a report of 2 pediatric cases. Respir Care. 2012;57:802-807. 66. Hoffstein V. Review of oral appliances for treatment of sleep-disordered breathing. Sleep Breath. 2007;11:1-22. 67. Schessl J, Rose E, Korinthenberg R, Henschen M. Severe obstructive sleep apnea alleviated by oral appliance in a three-year-old boy. Respiration. 2008;76:112-116. 68. Al-Samri M, Mitchell I, Drummond DS, Bjornson C. Tracheostomy in children: a population-based experience over 17 years. Pediatr Pulmonol. 2010;45:487-493.
research-article2014
CPJXXX10.1177/0009922814527501Clinical PediatricsBodenner et al
Article
Assessment and Treatment of Obstructive Sleep-Disordered Breathing
Clinical Pediatrics 2014, Vol. 53(6) 544–548 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0009922814527501 cpj.sagepub.com
Kaitlyn Anne Bodenner1, Supriya K. Jambhekar, MD2, Gulnur Com, MD2, and Wendy L. Ward, PhD2,3
Abstract Obstructive sleep-disordered breathing (OSDB) is a condition that affects 1% to 3% of the pediatric population. These disorders are difficult to diagnosis and left untreated may be serious, including not only medical comorbidities but also cognitive, academic, behavioral, and emotional sequelae. This article is designed to bring awareness of the severity and prevalence to family physicians and pediatricians. It reviews detailed information concerning OSDB, including the predisposing factors, assessment of presenting features, and treatment. Keywords obstructive sleep apnea, sleep-disordered breathing, pediatric sleep disorder
Introduction Obstructive sleep-disordered breathing (OSDB) is defined as obstruction of the upper airway during sleep that negatively affects sleep quality.1 “This may involve either complete upper airway obstruction (apnea), repeated episodes of partial upper airway obstruction (hypopnea), or prolonged increased upper airway resistance syndrome (UARS).”1(p65) OSDB is thought to be caused by “either anatomic narrowing of the upper airway, or increased collapsibility of UAW muscles, or abnormal neural control; usually a combination of all three events.”1(p67) Recently, the American Academy of Pediatrics2 released guidelines on the detection of OSDB in primary care settings, given findings that symptoms of OSDB may be missed, which can result in many adverse outcomes if left untreated. In addition, it has been shown that there is no correlation between symptoms and clinic findings and OSDB.3 Obstructive events during sleep are not easily identified in the general pediatrics setting and may result in many adverse outcomes if left untreated.4 In fact, given the range and severity of comorbidities associated with OSDB, it is essential that pediatric practitioners be aware of this condition and screen all children at well-child visits for the condition.5 Family physicians and pediatricians need to be aware of the disorder in children, the presenting features, the comorbidities, the predisposing factors, and the need for screening and referral
Why Obstructive Sleep-Disordered Breathing Is a Problem Obstructive sleep-disordered breathing affects approximately 1% to 3% of the pediatric population.6 With an estimated pediatric population of 74.2 million,7 there could be an estimated 1.5 to 2.2 million with OSDB in the United States. If untreated, OSDB results in a variety of negative outcomes. Medical complications include, impaired somatic growth, nocturnal enuresis, failure to thrive, and possible neuronal injury in the hippocampus and frontal cortex.8-11 OSDB in children has been linked to impaired cardiac function and structure,12 and other cardiovascular complications including impaired right ventricular function and cor pulmonale.13,14 Furthermore, OSDB has been associated with blood pressure dysregulation in some but not all studies.15 Circulating inflammatory mediators and increased insulin resistance have been posited as one of the potential process mechanisms, especially in obese children.16,17 OSDB may also be associated with extraesophageal reflux,18 lower exhaled
1
University of Central Arkansas, Conway, AR, USA University of Arkansas for Medical Sciences, Little Rock, AR, USA 3 Arkansas Children’s Hospital, Little Rock, AR, USA 2
Corresponding Author: Wendy L. Ward, Arkansas Children’s Hospital, 1 Children’s Way, Slot 512-21, Little Rock, AR 72202, USA.
545
Bodenner et al
Table 1. Questions Physicians Should Ask Patients or Caregivers for Assessment of Obstructive Sleep-Disordered Breathing.a Questions About Nighttime Manifestations Does he or she snore? Does he or she have uneven breathing? Does he or she have trouble breathing while sleeping? Have you ever witnessed them choke or make loud gasping sounds during the night? Is it difficult to awake your child? Does your child often wet the bed? How would you describe their sleeping position? (strange sleeping position) Does he or she sweat profusely while they are asleep? Is their sleep restless or do they startle awake?
Questions About Daytime Manifestations Do they awake with morning headaches or vomiting? Is he or she doing well in school? If not, has there been a recent onset of school complaints? Does he or she have attention problems or difficulty focusing? Has your child exhibited a depressed mood? Does the child have low appetite on wakening? Does he or she have memory problems? Have you noticed hyperactivity, irritable mood, or any other behavioral problems? Does he or she seem excessively tired during the daytime?
a
This table was adapted from one published in 2010.
nitric oxide/less eosinophilic and more neutrophilic type of asthma,19 and acutely decompensated heart failure.20 A causal relationship between nighttime sleep fragmentation and daytime neurobehavioral dysfunction has been suggested by multiple studies.9,21-24 Neurobehavioral dysfunctions associated with OSDB include poor school performance, cognitive dysfunction, inattentiveness, impaired memory, IQ deficit, executive function abnormalities, and so on.10,25-27 Behavioral outcomes are common as well—hyperactivity, conduct disorder, aggressive behavior, and so forth.25,26 Emotional outcomes are also present, for instance, quality-of-life impairment and depressive symptoms.28 Recent studies have also linked the neurobehavioral consequences of OSDB in children to inflammatory markers. However, these relationships are complex, as some circulating factors are associated with poor outcomes and others are protective.26,29,30
Predisposing Factors Enlarged tonsils and adenoids are the most common cause of OSDB in children.31-35 Obesity is another risk factor for OSDB.36,37 The reported prevalence of OSDB in obese youth ranges from 37% to 66%.38-40 Other conditions increasing the likelihood of OSDB are genetic syndromes, including Down syndrome and Prader–Willi syndrome; craniofacial anomalies, including syndromes such as Apert syndrome or Crouzon syndrome, achondroplasia, Arnold–Chiari malformation; neuromuscular syndromes, including muscular dystrophy; and cerebral palsy due to muscle tone abnormalities.
Assessment and Presenting Features Patients present a wide variety of nonspecific symptoms that make identification of the disorder challenging. The
American Academy of Pediatrics has presented guidelines on assessment of obstructive sleep apnea syndrome, a condition involving OSDB and impaired daytime functioning.41 This document suggests that pediatric patients may present with daytime manifestations of OSDB, including the cognitive, school, emotional, and behavioral comorbidities noted above. The symptoms include snoring, whistling noises, uneven/ jagged breathing while sleeping, occasional choking, and/or unusual sleep positions (often with the neck hyperextended). In addition, patients present with a myriad of sleep concerns—startling awake, restless sleep, profuse sweating. In the morning, they may have difficulty awakening or irritability on waking, morning headaches, vomiting, and lack of appetite for breakfast due to hypoventilation. Diagnosing OSDB first requires taking a detailed medical history, including daytime symptoms, nighttime symptoms, and adverse outcomes as described above (see Table 1) followed by physical examination. Radiological evaluation, including neck X-ray to look for enlarged adenoids, may help with diagnosis. Magnetic resonance imaging of the brain may be ordered in some patients to rule out foramen magnum stenosis or Arnold–Chiari malformation. X-ray cephalometry, computed tomography scanning and magnetic resonance imaging, dynamic scanning protocols (ultrafast computed tomography or cine magnetic resonance imaging), upper airway endoscopy during sleep, pressure measurements for the critical closing pressure are some other methods that may be used in selected patients.42-45 Further referral to a pediatric sleep center may be necessary for comprehensive assessment. Diagnosis is greatly supported by overnight polysomnography (PSG). PSGs assess “airflow at nose and mouth, thoracic and abdominal wall movements to detect obstructive respiratory events, oximetry for oxygenation, and end tidal CO2
546 to assess ventilation.”1(p6) Electroencephalography and electro-oculogram channels monitor stages of sleep throughout the study. Other conditions like periodic limb movements during sleep, nocturnal seizures, and parasomnias can also be detected on PSG. However, the PSG alone cannot be the sole assessment tool for several reasons. First, there are no proven norms for children and the exact significance of abnormal numbers in terms of predictive value for adverse outcome or need for treatment is not clearly defined. Second, significant neurobehavioral outcomes have been linked to clinical symptoms of OSDB without classical findings on PSG.46 Hence, diagnosis of OSDB and decision to treat are made based on a combination of all approaches.
Treatment of Obstructive Sleep-Disordered Breathing Once the diagnosis of OSDB is made and a decision to treat is taken, nasal steroids or leukotriene antagonists are helpful especially with upper airway congestion and mild adenoidal growth.47,48 It should be noted that while melatonin, antidepressants, and nonbenzodiazepine sleep-inducing medications may be used to help the patient sleep, they cannot be used as a sleep aid until the disordered breathing during sleep is improved and may in fact exacerbate OSDB. Tonsillo-adenoidectomy is usually the first line of treatment for children who have enlarged tonsils and adenoids and has been shown to help in a majority of these children.49 A wide range of additional surgical options (uvulopalatopharyngoplasty, maxillomandibular advancement, etc) are available as appropriate. However, a significant percentage of children continue to have OSDB following surgery50,51 or are poor candidates for surgery. This population includes mainly obese children49 and youth with craniofacial anomalies or neuromuscular difficulties.1 These children require treatment with positive airway pressure (PAP), either continuous positive airway pressure (CPAP) or bilevel positive airway pressure (B-PAP). CPAP has been shown to be effective in controlling the respiratory events and improving sleep parameters in children.53-58 “The main limitation of PAP use is poor adherence.”1(p68) Adherence studies in adults have shown that 30% to 46% patients with OSDB discontinue the use of PAP against medical advice.59,60 Emerging evidence regarding pediatric compliance with PAP treatment is available.53,54 It has been thought that early intensive intervention, including behavioral techniques such as systematic desensitization and reinforcement strategies will help in improving adherence with PAP use.53,59,61-63 Some patients describe difficulty initially tolerating their mask due to a claustrophobic reaction to the mask, significant sensory sensitivities, which
Clinical Pediatrics 53(6) may decrease tolerance to the feel of the equipment or the air pressure, or general unfamiliarity with the equipment, which may induce fear reactions. Children with developmental delays also have lowered adherence rates on average, as do individuals with disruptive behaviors.64 Structured adherence programs for PAP use are available and effective.65 Referral to a pediatric sleep center may be necessary for comprehensive management in some children. Oral appliances that attempt to increase upper airway size are used successfully in adults44,66; however, there is very little experience in children.67 The last option is tracheostomy, which is often effective but is considered highly invasive. In addition, tracheostomy is associated with increased morbidity and mortality.68
Conclusion In summary, OSDB is a common and often undetected condition that has a wide-ranging impact on patient functioning, including not only medical comorbidities but also cognitive, academic, behavioral, and emotional sequelae. Family physicians and pediatricians need training and education on sleep disorders, crucial in identification and treatment of pediatric patients with OSDB, to prevent these myriad health effects. Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
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