1. Obstructive Sleep Apnea in Children
Zafer Soultan,MD
Associate Professor of Pediatrics
Director, Pediatric Pulmonary and Sleep Medicine
SUNY, Upstate Medical University

2. Pediatric Obstructive Sleep Apnea
Primary care physicians, including pediatricians and family practitioners, are often the first healthcare professionals with the opportunity to recognize and diagnose pediatric obstructive sleep apnea (OSA). Many other medical practitioners, particularly otolaryngologists and pulmonologists, also see children with signs and symptoms of OSA. In addition, because the clinical manifestations of pediatric OSA include behavioral and academic problems, mental health professionals, including psychologists and teachers may encounter children whose primary problem is OSA.
The following presentation will provide an overview of OSA in children and adolescents. We will first describe the pathophysiology of pediatric OSA and its epidemiology in normal and high risk groups. This will be followed by a summary of clinical features and a discussion of diagnostic methods. The potentially serious consequences of pediatric OSA will be highlighted, followed by a discussion on treatment options currently available for pediatric OSA.

4. - 10 +1
Weak muscles

5. Cross-Section of Oropharynx
Nasal obstruction
Tonsillar hypertrophy
This is a sagittal section of the upper airway. Obstruction can occur from narrowing at any portion of the upper airway, for example choanal atresia, adenotonsillar tissue hypertrophy or micrognathia.
Large tongue
Micro- or retrognathia

6. Pathophysiology of OSA
Neuromotor tone
Cerebral palsy
Genetic diseases
Structural factors
Adenotonsillar hypertrophy
Craniofacial abnormality
Obesity
OSA
The etiology of pediatric OSA is not yet fully understood. It is thought to be due to a combination of structural, neuromotor and other factors. Structural factors result in upper airway narrowing; this may be due to adenotonsillar hypertrophy or craniofacial anomalies (e.g., micrognathia, midfacial hypoplasia). Obesity increases the risk for OSA by some combination of increasing the pharyngeal resistive load, increasing the compliance of the pharynx, or other mechanisms. Abnormal neuromotor tone is a prominent factor in some children with OSA, such as those with cerebral palsy. However, subtle abnormalities in upper airway neuromotor control are probably present in all patients with OSA - if not, these patients would obstruct during wakefulness as well as sleep. Subclinical neuromotor abnormalities may explain why some children with adenotonsillar hypertrophy develop OSA, whereas other children with similarly enlarged tonsils and adenoids do not. Other factors include a genetic predisposition, hormonal effects (particularly testosterone), and probably other, as yet undefined, factors.
A combination of structural, neuromotor and other factors are necessary for the development of OSA. However, the degree to which each factor contributes will differ. Thus, in a child with severe craniofacial anomalies, upper airway narrowing may predominate, whereas in a child with muscular dystrophy, decreased upper airway tone may predominate.
Other factors
Genetic
Hormonal ?

7. Risk Factors for Residual Obstructive Sleep Apnea After Adenotonsillectomy in Children
Imanguli M; Laryngoscope 2016

8. Sleep Disordered Breathing
Spectrum of conditions determined by relative amount of upper airway obstruction:
(CIRCLES DISPLAY INCREASING UPPER AIRWAY OBSTRUCTION)
PS - Primary snoring:
NOISY BREATHING
UARS - Upper airway resistance syndrome:
NOISY BREATHING + DISTURBED SLEEP
OH - Obstructive hypoventilation:
NOISY BREATHING ± DISTURBED SLEEP +  CO2 and/or  SaO2
OSA - Obstructive sleep apnea:
NOISY BREATHING ± DISTURBED SLEEP +  CO2 and/or  SaO2 + ABSENCE OF AIRFLOW
Sleep disordered breathing occurs as a syndrome that constitutes a spectrum of conditions of which pediatric obstructive sleep apnea is the severest. The primary determinant of each condition is the amount of upper airway obstruction (UAO) that occurs during sleep. At one end of the spectrum without known consequences is primary snoring, resulting from UAO severe enough to produce noisy breathing alone. Presence of symptoms of sleep disruption and/or physiologic alterations in gas exchange along with the amount of airflow reduction across the upper airway are additional determinants for other conditions across the spectrum namely UARS, obstructive hypoventilation and OSA.

9. OSA Epidemiology Habitual snoring 1.5-28% OSA – 1% to 5%
M:F ratio approximately equal
Prevalence is higher among African Americans, obese, AT hypertrophy, craniofacial anomalies

10. Symptoms of OSA Frequent snoring (≥3 nights/wk) Labored breathing
Gasps/snorting noises
Apnea
Enuresis (especially secondary)
Sleeping in a seated position or with the neck hyperextended
Cyanosis
Headaches on awakening
Daytime sleepiness
Attention-deficit/hyperactivity disorder
Learning problems

11. Signs of OSA Underweight or overweight Tonsillar hypertrophy
Adenoidal facies
Micrognathia/retrognathia
High-arched palate
Failure to thrive
Hypertension

13. Sequelae

14. Neuropsychological and Cognitive Problems
Deficits in:
Learning, memory, and visuospatial skills
Language, verbal fluency, and phonological skills
Concept formation, analytic thinking, and verbal and nonverbal comprehension
School performance and mathematical abilities
Executive functions; mental flexibility, impulse control and working memory
There is correlation between different polysomnographic factors and cognitive outcomes.

15. Behavioral abnormalities
Hyperactivity; in younger kids
ADHD
Hypersomnolence
Somatization
Depression
Atypicality
Aggression, and abnormal social behaviors

16. Improvement after treatment
The majority of investigations demonstrated agreement about post treatment improvement of behavior, quality of life (QoL), hyperactivity, ADHD, and impulsivity
Additional studies have demonstrated improved cognitive function (by using objective measurement) after treatment of OSAS, including measures of general intelligence, attention, memory, and analytic thinking

17. Gozal, D. Pediatrics 1998;102:
Copyright ©1998 American Academy of Pediatrics 17

18. Neurocognitive deficits were found at baseline in SDB children compared to controls; 10 point IQ difference (P<.001) and similar deficits in language and executive function.
Adenotonsillectomy improved respiratory parameters and snoring frequency at 6 months post surgery, neurocognitive performance did not improve relative to controls. Kohler MJ, PLoS ONE. 2009;4(10):e7343
Phonologic processes and verbal fluency did not improve to normal. Montgomery-Downs Eur Respir J. 2005;25(2): 336–342. Lundeborg I, Clin Linguist Phon. 2009;23(10):751–761

20. Conclusions by the experts
The majority of these studies suggest that in developing children who are dependent on executive function, cognition, and behavioral skills important for daily function and school performance, treatment of childhood SDB has benefits.

21. Cardiovascular dysfunction
Subclinical left sided and right sided and biventricular cardiac dysfunction
Correlation between the presence and severity of OSA and elevated systolic and diastolic BP
Systemic hypertension may occur in children with OSA. In this slide, the diastolic blood pressure index (the difference between the subject’s mean blood pressure during sleep and the blood pressure at the 95th percentile for age and height) is plotted against the apnea index. There is a significant correlation between the blood pressure index and apnea index.49
49. Marcus CL, Greene MG, Carroll JL. Blood pressure in children with obstructive sleep apnea. Am J Respir Crit Care Med 1998;157(4 Pt 1):

22. Effect on growth Girls Boys
Failure to thrive (FTT) as a consequence of pediatric OSA has been documented in earlier reports Presentation of OSA in this manner has become less common, probably due to earlier recognition and treatment of the condition. In a small study of children with OSA and FTT catch-up growth was noted after adenotonsillectomy in all children. Adenotonsillectomy has also been shown to increase the height and weight velocities of children with normal growth and OSA. This is also true of obese children who undergo adenotonsillectomy for OSA. The mechanisms of failure to thrive in children with OSA are not completely understood but are likely related to an increase in night-time energy expenditure caused by increased respiratory effort and also due to likely disruption of the growth hormone-IGF 1 pathway.
This figure shows the improvement in weight percentile following adenotonsillectomy in a group of non-obese children with OSA. Girls are shown in panel A, boys in panel B.41 Note that these children had relatively mild OSA, with a mean obstructive apnea index of 6/hr, and SaO2 nadir of 85%. There was a decrease in work of breathing postoperatively, but no change in caloric intake.
37. Ahlqvist-Rastad J, Hultcrantz E, Melander H, Svanholm H. Body growth in relation to tonsillar enlargement and tonsillectomy. Int J Pediatr Otorhinolaryngol 1992;24(1):55-61.
38. Bar A, Tarasiuk A, Segev Y, Phillip M, Tal A. The effect of adenotonsillectomy on serum insulin-like growth factor-I and growth in children with obstructive sleep apnea syndrome. J Pediatr 1999;135(1):76-80.
39. Everett AD, Koch WC, Saulsbury FT. Failure to thrive due to obstructive sleep apnea. Clin Pediatr (Phila) 1987;26(2):90-2.
40. Freezer NJ, Bucens IK, Robertson CF. Obstructive sleep apnoea presenting as failure to thrive in infancy. J Paediatr Child Health 1995;31(3):172-5.
41. Marcus CL, Carroll JL, Koerner CB, Hamer A, Lutz J, Loughlin GM. Determinants of growth in children with the obstructive sleep apnea syndrome. J Pediatr 1994;125(4):
Girls
Boys
Marcus et al. J Pediatr 1994

23. Soultan, Z. Arch Pediatr Adolesc Med. 1999;153:33-37.

24. Soultan, z. Arch Pediatr Adolesc Med. 1999;153:33-37.

25. Inflammation
Log CRP levels (mean ± SD) for 81 children with AHI of <1 (control), AHI of ≥1 and <5 (mild SDB), or AHI of ≥5 (SDB). Log CRP levels in the SDB group differed from those in the mild SDB and control groups (P < and P = .04, respectively).

26. Individual serum C-reactive protein (CRP) levels in 20 children with sleep-disordered breathing before (Pre) and after (Post) adenotonsillectomy.
Gozal L, J Clin Sleep Med. Author manuscript; available in PMC 2007 Apr 4.

27. Inflammation
Increased glucocorticoid and leukotriene receptors in adenotonsillar tissue from children who had OSAS compared with tissue from children who experienced chronic throat infections. Khalyfa A et al, Goldbart AD et al

28. Diagnosis; just ask and verify

29. Sensitivity %
Specificity 72-87%

30. Diagnosis Nocturnal oximetry
Sensitivity 67%, and specificity 60% for moderate OSA AHI > 5. Kirk et al.
Good correlation with PSG, but 80% of the 223 children had normal, inconclusive, or technically unsatisfactory oximetry. Brouillette et al
Home sleep study; abbreviated form. Promising for school-aged children and for AHI >5

31. Pediatric Polysomnography
EEG
EOG
Nasal EtCO2
Nasal Oral Airflow
Chin EMG (2)
Microphone
Sao2
EKG
Tech Observer
Video Camera
This illustration shows the standard types of monitoring devices used in pediatric polysomnographic evaluation in most sleep laboratories. Electroencephalography (EEG), electro-oculography (EOG) and submental electromyography (EMG) are used to monitor sleep architecture and arousals. The presence of airflow is assessed by nasal/oral thermistors and/or capnography and/or pressure transducers. Chest wall excursion is usually measured by means of inductance plethysmography or strain gauges. Intercostal EMG can be used to measure respiratory effort, particularly paradoxical chest wall movement. A pulse oximeter measures oxygenation. CO2 is generally assessed either by means of a transcutaneous CO2 monitor or an end-tidal CO2 probe; this is helpful to assess hypoventilation. The entire procedure is observed by a technician who documents arousals, parasomnias, abnormal sleeping position, and attends to any technical problem. Esophageal pH may be monitored if gastroesophageal reflux is a clinical consideration.
Children usually require advance preparation, which may include a tour of the sleep lab prior to the polysomnographic evaluation. It generally requires more time and patience to set up a child for a polysomnogram, and it is usually helpful if a parent is present during the study. The sleeping room should be child-friendly, and technicians in the sleep lab should be comfortable and familiar working with children.30
30. Zaremba EK, Barkey ME, Mesa C, Sanniti K, Rosen CL. Making polysomnography more "child friendly:" A family-centered care approach. Journal of Clinical Sleep Medicine 2005;1(2):
Respiratory Effort
Documents arousals, parasomnias, abnormal sleeping position, and attends to any technical problem
Leg EMG (2)
Record behavior
Courtesy of Dr. Carol Rosen

32. Obstructive Sleep Apnea
OBSTRUCTIVE APNEA
OBSTRUCTIVE APNEA
Two patterns of obstruction occur in children with OSAS. This tracing depicts cyclic obstructive apneas, similar to that seen in adults. There are several episodes of obstructive apnea (evidenced by lack of airflow on both the oronasal (NAF) and pCO2 channels) despite continued respiratory effort. This is associated with oxyhemoglobin desaturation to 81%.
HYPOXEMIA FOLLOWING APNEIC EPISODE

33. Obstructive Hypoventilation
PARADOXICAL RIB-CAGE MOTION
This tracing depicts obstructive hypoventilation, i.e. persistent partial upper airway obstruction associated with hypercapnia. The child, who was snoring at the time, has paradoxical inward rib cage motion. There are no obstructions - airflow can be seen for each respiratory effort. The partial obstruction has resulted in severe hypoxemia (SaO2 in the 60s) and hypercapnia (end-tidal pCO2 in the 70s). This pattern can occur for prolonged periods of time without arousal from sleep.
HYPERCAPNIA

34. Pediatric Polysomnography
Role of Polysomnography
Differentiate OSA from primary snoring
Define severity of OSAS
Differential diagnosis
Evaluate success of treatment
The ICSD 221 diagnostic criteria for pediatric OSA incorporates polysomnographic criteria as well, making pediatric polysomnography essential for diagnosing pediatric OSA. Additionally, polysomnography is performed to confirm the diagnosis of obstructive sleep apnea when it is suspected on clinical grounds, and to differentiate between OSA and primary snoring. Polysomnography helps to delineate the severity of the OSA, since this will affect the choice of treatment and degree of postoperative monitoring. Polysomnography should be considered to evaluate symptoms such as excessive daytime sleepiness or failure to thrive. Finally, a polysomnogram may be necessary to evaluate the success of an intervention, such as adenotonsillectomy, particularly in a situation in which post-treatment residual symptoms or risk factors for OSA exist, or when OSA was severe prior to treatment.
21. American Academy of Sleep Medicine. International classification of sleep disorders. 2 ed. Westchester, IL: American Academy of Sleep Medicine; 2005. 34

35. Treatments: Adenotonsilletcomy, Partial tonsillectomy
Pain and poor oral intake
Bleeding, infection, anesthetic complications, respiratory decompensation, velopharyngeal incompetence, subglottic stenosis, obesity, and, rarely, death.
In-patient observation: age <3 years, severe OSAS, presence of cardiac complications, failure to thrive, obesity, and presence of upper respiratory tract infection (URI).

36. Does it work?

39. Persistence of OSA after AT
OSA (AHI >1) persistence 19%-73%, AHI ≥5/hour 13%-29%.
Obesity: 51% AHI ≥5/hour; OR of persistent OSAS in 3.21 to 4.7.
Severe OSA
Very young and older children > 7 years independent of obesity
Coexisting asthma, and family history
Likely in children with craniofacial anomalies, Down syndrome, and neuromuscular disease

40. Cure, improve with residual OSA after AT
AHI: Pre AT 21.4 (2.3–147.5), post 1.5 (0–20.0) p <.001
The prevalence of residual OSA for AHI >1 criteria (72%), and
AHI >2 was 38%
Retrospective review of chart of 169 patients (all ages) undergone AT for treatment of OSA- PSG before (median 78 days) and after (median 114 days)
Risk Factors for Residual Obstructive Sleep Apnea After Adenotonsillectomy in Children
Imanguli M; Laryngoscope 2016

41. Conclusions by the experts
AT is the most effective surgical therapy for pediatric patients, leading to an improvement in polysomnographic parameters in the vast majority of patients
Significant proportion of patients are left with persistent OSAS after AT.
13-29% in low-risk population for AHI ≥5/hour
73% when including obese children and adolescents and a conservative AHI ≥1/hour.
Children at highest risk of persistent OSAS are those who are obese and those with a high preoperative AHI, especially those with an AHI ≥20/hour, as well as children >7 years of age.

42. This is a 15 month old boy sleeping quietly wearing his nasal CPAP mask.

43. Positive Airway Pressure
This slide depicts the therapeutic effect of continuous positive airway pressure (CPAP). In the panel on the left, you can see upper airway closure in an untreated sleep apnea patient. Note that the airway closure is diffuse, involving both the palate and the base of tongue. In the second panel, CPAP is applied and the airway is splinted open by the positive pressure.

44. Positive airway pressure
Indications: persistent OSAS after AT, usually in obesity or craniofacial anomalies, and when surgery is contraindicated
Effective in resolving symptoms and polysomnographic abnormalities
Adherence?

45. Special Considerations for CPAP in Children
Need wide variety of mask sizes and styles to fit children
Compliance may be enhanced by behavioral techniques
Empowerment
Positive reinforcement
Desensitization
Role modeling
Children require special care and consideration for successful treatment with CPAP. A wide variety of mask sizes and styles are required to insure the proper fit. Compliance rates for CPAP use in children range from 50% to 100%.55 Certain behavioral techniques can enhance compliance. These may include: negotiating a contract with the child about CPAP use and allowing the child to have a say in establishing parameters for use; training parents to manage resistant behavior and to use positive reinforcement; the use of desensitization and shaping techniques which gradually expose the child to increasingly longer periods of use and higher CPAP pressures over several weeks; and providing appropriate peer role models.56
Careful attention to minor, but sometimes troublesome side effects can enhance compliance. These include nasal congestion, nasal dryness and rhinorrhea, eye irritation, and chemical conjunctivitis, facial dermatitis, and an increase in oral secretions. Nasal sprays can be used to treat nasal obstruction and rhinitis, and a chin strap can help minimize mouth breathing. A prolonged ramp time can be used to allow the child to acclimate to the prescribed pressure. Humidification of the inspired air may be helpful.
55. Marcus CL, Ward SL, Mallory GB, et al. Use of nasal continuous positive airway pressure as treatment of childhood obstructive sleep apnea. J Pediatr 1995;127(1):88-94.
56. Koontz KL, Silfer KJ, Cataldo MD, Marcus CL. Improving pediatric compliance with positive airway pressure therapy: the impact of behavioral intervention. Sleep 2003;26(8):

46. Medications
Intranasal steroids may be considered for treatment of mild OSA; AHI <5/hour

47. Sleep Med. 2007 Mar;8(2):128-34. Epub 2007 Jan 18.
Rapid maxillary expansion in children with obstructive sleep apnea syndrome: 12-month follow-up.
Villa et all

48. The Childhood Adenotonsillectomy Trial (CHAT)

50. Conclusions by the CHAT
As compared with a strategy of watchful waiting, surgical treatment for the obstructive sleep apnea syndrome in school-age children did not significantly improve attention or executive function as measured by neuropsychological testing but did reduce symptoms and improve secondary outcomes of behavior, quality of life, and polysomnographic findings, thus providing evidence of beneficial effects of early adenotonsillectomy. (Funded by the National Institutes of Health; CHAT ClinicalTrials.gov number, NCT )
Nothing is a medicine too (normalization of polysomnographic findings in 46% (79% after AT), but only for certain patients!
“Normalization of polysomnographic findings in a large number of children in the watchful-waiting group and an absence of significant cognitive decline in this group indicate that medical management and reassessment after a period of observation may be a valid therapeutic option”
After a 7-month interven- tion period, school-age children with the ob- structive sleep apnea syndrome without pro- longed oxyhemoglobin desaturation who under- went surgery did not have significantly greater improvement in attention and executive function, as measured by means of neuropsychological testing, than did children in the watchful-waiting group. However, surgery resulted in greater re- ductions in symptoms and greater improvements in behavior, quality of life, and polysomnographic findings, with effect sizes in the moderate-to- large range. Polysomnographic findings were normalized in the majority of children (79%) in the early-adenotonsillectomy group, although poly- somnographic abnormalities also resolved in 46% of the children randomly assigned to watchful waiting

51. Send to:
Pediatrics Jun;59(6):
Hypoventilation during sleep in children who have lymphoid airway obstruction treated by nasopharyngeal tube and T and A.
Kravath RE, Pollak CP, Borowiecki B.
Abstract
Three children with sleep apnea, alveolar hypoventilation, apparent mental retardation, and poor growth associated with chronically enlarged tonsils and adenoids were treated with the use of a nasopharyngeal tube followed by tonsillectomy and adenoidectomy. The effectiveness of this therapy was documented by polygraphic recording of sleep stages and respirations, and by correlation with serial arterial blood gases and pH. The nasopharyngeal tube was well tolerated, easy to use, and effective in diagnosis and treatment. We suggest that its use be further evaluated in patients with obstructive apnea.
PMID: [PubMed - indexed for MEDLINE]

53. Kravath’ method

54. We end with the AAP recommendations
Inquire about snoring, and if positive do thorough evaluation.
Polysomnogram (PSG), or referral to a sleep specialist or otolaryngologist
If PSG is not available; nocturnal video recording, nocturnal oximetry, daytime nap PSG, or ambulatory PSG.
Adenotonsillectomy (AT)
Monitor high-risk patients after AT
Reassess all patients with OSAS for persisting signs and symptoms after therapy
Reevaluate high-risk patients for persistent OSAS after AT
CPAP
Weight loss

55. AAP recommendations cont.
Topical intranasal corticosteroids for children with mild OSAS in whom adenotonsillectomy is contraindicated or for children with mild post-operative OSAS
Not AAP recommendation, do nothing?

56. Conclusion
We should not overlook OSA, because it does have serious consequences
Early diagnosis and treatment of may improve a child’s long-term cognitive and social potential and school performance.

57. The source and useful resource
Diagnosis and Management of Childhood Obstructive Sleep Apnea Syndrome Carole L. Marcus, Lee Jay Brooks, Kari A. Draper, David Gozal, Ann Carol Halbower, Jacqueline Jones, Michael S. Schechter, Stephen Howard Sheldon, Karen Spruyt, Sally Davidson Ward, Christopher Lehmann and Richard N. Shiffman Pediatrics 2012;130;576; originally published online August 27, 2012; DOI: /peds ; The online version of this article, along with updated information and services, is located on the World Wide Web at: