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ENT Perspectives on Pediatric OSA
Ron B. Mitchell, MDProfessor and Vice Chairman,
Department of Otolaryngology- Head and Neck Surgery Chief of Pediatric OtolaryngologyUT Southwestern Medical CenterChildren's Medical Center Dallas
Objectives:} To discuss:
} The evidence that surgical therapy for OSA in children leads to good outcomes
} Future research needs and opportunities for collaboration between otolaryngologists and orthodontists
• 12 Pediatric Otolaryngologists• 7 Pediatric Sleep Medicine specialists• 2000 T&A• 3000 Pediatric PSG with 80% <12• Multidisciplinary clinics and conferences
Why should ENTs and Orthodontists be interested in mouth breathing/ snoring/OSA in Children?
• general well-being• emotional well-being• behavioral health• Impact on the family
Why should ENTs and Orthodontists be interested in mouth breathing/ snoring/OSA in Children?
OSA in Children
• > 500,000 T&A procedures (15% of pediatric surgery) are performed per year in children in the USA
• Majority of procedures for OSA• Average age 4-6 years• 25% of 4-6 year-olds are habitual snorers/ mouth breathers
• T&A is the most common major surgical procedure in children
• 1-4% of children have OSA
• In obese/ DS children OSA >50%
What is the difference between Tonsillectomy and Tonsillotomy?
Tonsillectomy Tonsillotomy
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What is the difference between Tonsillectomy and Tonsillotomy?
Tonsillotomy
The rationale is that the remaining tonsillar capsule would form a biological dressing resulting in reduced postoperative morbidity
Dissection is between tonsillar capsule and superior constrictor/ Larger vessels are dissected and cauterized
Tonsillectomy
Tonsillotomy: Is it superior to Tonsillectomy
• > 200 published articles in the last 10 years- mostly small case series
• There are 3 systematic reviews of high-level studies that compare tonsillotomy to tonsillectomy:
• Fewer days before resolution of pain with tonsillotomy, at a mean 5.0 versus 7.6 days (P =.045)
• Secondary hemorrhage rates were lower with tonsillotomy 0.72% versus 3.56% (P = .04)
• long-term outcomes in tonsillotomy are important in determining rates of regrowth, which is the greatest detractor in otolaryngologists integrating this technique more widely
W alton J et al. System atic review of random ized controlled trials com paring intracapsular tonsillectom y w ith total tonsillectom y in a pediatric population. Arch O tolaryngol H ead N eck Surg. 2012;138:243-249.
Tonsillotomy: Is it superior to Total Tonsillectomy (TT)? What do I do?
• 80% tonsillectomy• Tonsillotomy when specifically requested by caregivers• Tonsillotomy in <2 year-olds• More tonsillotomy now than 5 years ago• Never had a bleed following tonsillotomy that went back to the OR; several
admissions for pain control/ dehydration/ bleeding that stopped without intervention
Clinical Practice Guideline (Update): Tonsillectomy in Children- Chair: Ron B Mitchel, MDUpdate to 2011 GuidelineTo be published in 2019
When do we need to get polysomnography (sleep study) in children?
• Symptoms and signs of OSA For >3 months + large tonsils = T&AWho do we want PSG on?
• Children under 2 • Children with significant comorbidities
• Obesity• Down syndrome / craniofacial abnormalities• Neuromuscular disorders• Sickle Cell disease• Mucopolysaccharidoses
OSA and Quality-of-Life in Children
• Most T&As in children are performed for OSA• The MAIN indication is quality-of-life
• OSA is associated with behavioral problems and is known to significantly affect quality-of-life
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What is the impact of OSA on quality-of-life in children?
} QOL of children with OSA is significantly worse than those of healthy normal children
} QOL of children with OSA were similar to children with juvenile rheumatoid arthritis or asthma
Stewart MG. Quality of life and health status in pediatric tonsil and adenoid disease. Arch Otolaryngol Head Neck Surg. 2000 Jan;126(1):45-8Georgalas C. Measuring quality of life in children with adenotonsillar disease with the Child Health Questionnaire: a first U.K. study. Laryngoscope. 2004 Oct;114(10):1849-55
• T&A has beneficial cardiovascular effects in children with OSA
• Children with cardiac morbidity associated with OSA should be treated early with T&A to reverse potentially serious cardiovascular sequelae
• There is a paucity of well-designed studies on this subject and a need to standardize the diagnosis of OSA with pre- and postoperative PSG and to identify the cardiac parameters that are affected by OSA in children
OSA: Down Syndrome
• 37 children with DS:• average age of 3.5 years• 67% had an abnormal AHI (>2) after T&A
• 11 children with DS• average age of 8.4 years• 82% had an abnormal AHI (>2) after T&A
• 84 children with DS (2018 UTSW unpublished)• average age of 7.2 years• 78% had an abnormal AHI (>2) after T&A
Merrell JA, Shott SR. OSAS in Down syndrome: T&A versus T&A plus lateral pharyngoplasty. Int J Pediatr Otorhinolaryngol 2007; 71:1197–1203.Shete MM, Stocks RM, Sebelik ME, Schoumacher RA. Effects of adenotonsillectomy on PSG patterns in Down syndrome children with obstructive sleep apnea: a comparative study with children without Down syndrome. Int J Pediatr Otorhinolaryngol 2010; 74:241–244.
Pediatric OSA and Down SyndromeChildhood Obesity
• In the United States, the percentage of children and adolescents affected by obesity has more than tripled since the 1970s
• Childhood obesity is an international problem
• 1 in 3 school age children and young people (6 to 19 years) in the United States is overweight/obese
OSA and Obesity in Children
• The incidence of OSA in children with obesity ranges from 13 –50% or 10x that of normal weight children• Otolaryngologists are more likely to see obese children with OSA
for possible T&A
Everyone in our family snores!!!
OSA and Obesity in Children
• Why is OSA more common in obese children?
• Adipose tissue deposited around the pharynx or subcutaneous tissue of the neck leads to compression and a reduction in its cross-sectional area• Adipose tissue is deposited in the tongue itself• Adenotonsillar tissue further narrows the airway
• 30% of asymptomatic obese children have moderate to severe OSA
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• Adolescent obese children are at the highest risk of severe OSA
OSA and Obesity in Children-Treatment
• Weight reduction• CPAP- in addition or as an alternative to surgery• T&A• Other procedures- nasal, palate, tongue base, tracheostomy• Multidisciplinary approach
Outcome of T&A for Pediatric OSA
} Improvements in sleep parameters as measured by PSG
} Improvements in quality-of-life
} Improvements in behavior
} Improvements in healthcare utilizationFrom making the cure of a diseasemore grievous than its enduranceGOOD LORD DELIVER US
- Sir Robert Hutchinson, 1871-1960
T&A for OSA in Children
• Mitchell (2006) – 79 healthy children with OSA were studied with pre- and postoperative PSG• In all children AHI improved after surgery• All children with a pre-operative AHI <10 normalized after
surgery• 73% of children with AHI >10 normalized after surgery• In all children with persistent OSA caregivers reported snoring
at 3-month f/u
Mitchell RB. Adenotonsillectomy for obstructive sleep apnea in children: outcome evaluated by pre- and postoperative polysomnography. Laryngoscope. 2007 Oct;117(10):1844-54.
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1 5 9 1 3 1 7 2 1 2 5 2 9 3 3 3 7 4 1 4 5 4 9 5 3 5 7 6 1 6 5 6 9 7 3 7 7
OAHI
Children Ranked by Preoperative Severity of OSAS
Change in OAHI after Adenotonsillectomy
P re op er ativ e
How effective is T&A at improving QOL in children with OSA?
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S 1 S 2 S 3 S 4 P 1 P 2 P 3 P 4 E 1 E 2 E 3 D 1 D 2 D 3 C 1 C 2 C 3 C 4
OSA
-18
Mea
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tal S
core
P re -s u rg e ry M e a n
SleepDisturbanc
Physical Suffering
Emotional Distress
Daytime Problems
Caregiver Concerns
Mitchell RB. Quality of life after T&A for obstructive sleep apnea in children. Arch Otolaryngol Head Neck Surg. 2004 Feb;130(2):190-4.
• 60 children with PSG-proven OSA completed the OSA-18 QOL survey before and 3-months after tonsillectomy
•The changes in total score, in the scores for each domain of the OSA-18 survey were highly significant
Pediatric OSA and Behavior} Several large studies have shown that children with OSA have
behavioral problems:} Attention} Hyperactivity} Aggression} Irritability} Emotional and peer problems} Somatic complaints
} 20-30% of children with OSA score in the abnormal range using standardized behavioral instruments
} 5-10% continue to score in the abnormal range after T&A
Mitchell RB, Kelly J. Long-Term Changes in the Behavior of Children after Adenotonsillectomy for Obstructive Sleep Apnea. Otolaryngol Head Neck Surg. 2006 Mar;134(3):374-8 PMID 16500430
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Health Care Utilization in Children with OSA
} Children with OSA had significantly more hospital and outpatient visits and more use of antibiotics resulting in a 215% elevated healthcare use compared to controls
} Following T&A total healthcare costs were reduced by onethird and this change was not seen in the control or untreated group
} Most of the decreased morbidity was due to a reduction in upper respiratory tract infections
Tarasiuk A: Elevated morbidity and health care use in children with obstructive sleep apnea syndrome. Am J Respir Crit Care Med. 2007 Jan 1;175(1):55-61
Health Care Utilization in Children with OSA
• 4276 Children ages 2-16 years who had a diagnosis of adenotonsillar hypertrophy were studied. The main outcome was direct total
• Costs 2 years before and after T&A were compared
• After T&A, median costs during a 2-year period were reduced by 37%
• A reduction in drug costs was the main driver for cost reduction, and this was mostly for reduced costs of antibiotics by 66% and outpatient visits by 32%
Chang JJ, Mitchell RB. Cost Analysis of Tonsillectomy in Children Using Medicaid Data. J Pediatr. 2014 Jun;164(6):1346-51. PMID: 24631119
The ChildHood AdenoTonsillectomy study (CHAT)
• A Randomized Controlled Study of Adenotonsillectomy for Childhood Sleep Apnea supported by the NIH/ NHLBI
• Establishes causality
• 460 children enrolled - study completed March 2012
• Primary paper published in NEJM May 2013
The Childhood Adenotonsillectomy Trial (CHAT). N Engl J Med. 2013 Jun 20;368(25):2366-76
CHAT Study
RandomizationBaseline examNeuropsychevaluationQOL survey
230WWSC
230T&A
7MBaseline exam
PSGBehavior
QOL survey
460Children
WithPSG-proven
OSA
Cincinnati Children�s HospitalUniversity of Cincinnati
Children�s Hospital of PhiladelphiaUniversity of PennsylvaniaRainbow Babies and Children�s HospitalCase Western Reserve University
Cardinal Glennon Children�s Medical CenterSaint Louis University
Montefiore Children�s HospitalAlbert Einstein Medical Center
Children�s Hospital of BostonHarvard Medical School
The Childhood Adenotonsillectomy Trial (CHAT). N Engl J Med. 2013 Jun 20;368(25):2366-76
Inclusion CriteriaAges 5 to 9 years AHI ≥ 2 candidate for T&AExclusion CriteriaSignificant comorbiditiesSevere OSA Morbid obesity
1244 children considered candidates for T&A were screened with polysomnography
594 (48%) had an AHI ≥ 2
460 were randomized to T&A or WWSC
Mean AHI=4.750% were
overweight/ obese
CHAT Study
Conclusions:• Of all children
considered candidates for T&A by otolaryngologists >50% have a normal PSG
• Of children with OSA most have mild-moderate OSA
43 (3%) were excluded because of
severe OSA
The Childhood Adenotonsillectomy Trial (CHAT). N Engl J Med. 2013 Jun 20;368(25):2366-76
“Normalization” of OSA
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By Weight
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Obese
NormalWeight
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85
• AHI �normalizes� over 7 months in 46% of
children without T&A
• AHI �normalizes� over 7 months in 79% of
children with T&A
• AHI �normalizes� over 7 months in 29% of
obese children without T&A and in 68% of
obese children after T&A
The Childhood Adenotonsillectomy Trial (CHAT). N Engl J Med. 2013 Jun 20;368(25):2366-76
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OSA and symptom resolution over 7-months T&A vs. Non-T&A
Of 230 in Observation group 194 completed the
study
20 (12%) showed PSG and symptom
resolution
Of 230 in T&A group 185 completed the
study
133 (72%) showed PSG and symptom
resolution
Predictors of resolution:• low AHI• Not obese• Not African American
Conclusions:• T&A is highly effective at
normalizing AHI and symptom resolution
• Observation is less effective at normalizing AHI and poor at symptom resolution
CONCLUSIONS
} OSA is under-diagnosed and treated in children and is associated with behavioral problems and reduced quality-of-life in children
} T&A is associated with improvements in behavior, quality-of-life, healthcare utilization as well as sleep in children
} Results from CHAT has shown that T&A is more effective than observation at symptom relief in children with OSA
Pediatric Adenotonsillectomy Trial for Snoring (PATS)
• Study started in June 2016- >260 children enrolled• Study will randomize 460 children (3 to 12) with normal PSG (no “OSA”) to T&A
compared to observation• 12 months follow-up• Behavior, symptoms, quality of life and healthcare utilization will be studied
Why should ENTs and Orthodontists be interested in mouth breathing/ snoring/OSA in Children?
• If OSA =T&A= Resolution- What is the problem?
• Not all children who undergo T&A have resolution of OSA
• If children have mild OSA/ Snoring- often observation not T&A is recommended- is this correct or evidence based?
• Does pediatric OSA lead to adult OSA?
• Is the real problem nasal obstruction that is untreated and leads to OSA?
20%
40%
60%
80%
100%
Black NotBlack
Obese NonObese
MildOSA
71%
88%
67%
88% 86%
Cure Rate
N=103 N= 91 N=68 N=126 N=194
1913
Case 1:
• 8 yo boy referred by his orthodontist for “large tonsils” and snoring
• 2 year+ history of nightly snoring, occasional mouth breathing, no witnessed apneas
• Good student, not obviously tired during the day, does not nap during the day
• No significant PMH; no medications; tried a nasal steroid spray without improvement
• PSG- AHI=0.7 without hypoxemia or hypercapnia
• Orthodontist recommended T&A prior to starting orthodontics
• Would this child benefit from T&A?
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Nasal Obstruction and facial Growth
• There may be an association between nasal obstruction and poor facial growth• 20% of normal-weight children with OSA continue to have persistent OSA with
symptoms after T&A• ? many years of nasal obstruction/ OSA affects facial growth that is the cause of
persistent OSA after T&A• Equally, nasopharyngeal/ oropharyngeal crowding may be incidental to OSA and
does not improve after T&A
The problem with correlations:
Adenoid Facies• The features of the classical adenoid facies are:• 1. Long face: Entire face is elongated, especially the lower
one-third• 2. Lips: Short upper lip and larger lower lip • 3. Open mouth posture
• Vertical growth• Long face• Retruded lower
jaw• Crowded teeth• Poor airway/
breathing problems/ OSA
• TMJ problems
Mechanism for Craniofacial Change• Open mouth posture/mouth breathing occurs when the nasal resistance rises 2-3x
normal
• Secondary effects : . • The lack of tooth contact =excessive eruption of the posterior molar teeth=down and
back rotation of the mandible=open bite deformity. • To maintain an oral airway, the tongue assumes a low posture= loss of lateral forces on
the growing maxilla=narrow hard palate and cross-bite deformities
• Should children with upper airway obstructive symptoms and these facial characteristics be considered for a T&A?
• Has intervention resulted in resolution of altered facial characteristics for some patients?
Open bite
Cross bite
Narrow maxillary arch
Facial / adenotonsillar growth• By age 4 – 60% of
craniofacial skeleton has reached adult size
• By age 8- 80% of craniofacial skeleton has reached adult size
• By age 12 – 90% of craniofacial skeleton has reached adult size
Research/ Evidence
Experiments on the interaction between orofacial function and morphology. EarNoseThroatJ. 1987; 66, 201–208. Craniofacial morphology and airflow in children with primary snoring Eur Rev Med Pharmacol Sci. 2016 Oct;20(19):3965-3971.Changes in dentofacial morphology after adenotonsillectomy in young children with obstructive sleep apnoea--a 5-year follow-up study. J Orthod2006; 28: 319-326.Midfacial and Dental Changes Associated with Nasal Positive Airway Pressure in Children with Obstructive Sleep Apnea and Craniofacial Conditions J Clin Sleep Med. 2016 Apr 15;12(4):469-75.
• Between 1970 and 1980, a number of experiments on newborn rhesus monkeys were performed- a small silicone head was placed within the nostrils of infant monkeys for the first 6-months of life. This led to:• narrowing of dental arches• decrease in maxillary arch length• anterior cross bite• maxillary overjet• increase in anterior facial height• systematic changes in the oral-facial muscles (hypotonia)
• In children with primary snoring there was an association between the total facial vertical divergence ( the Frankfurt mandibular Angle- FMA) and the severity of nasal obstruction measured by rhinomanometry
• Some degree of normalization of the growth pattern after T&A in children with OSA
• PAP use in compliant versus non-compliant children may alter normal facial growth
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Research/ Evidence
• Swedish investigators suggested that:mouth breathing=inflammatory reaction in the tonsils= tonsillar hypertrophy
• Rapid maxillary expansion (RME)- SR of 17 studies with 314 children (7.6 ± 2.0 years old) with high-arched and/or narrow hard palates and OSA• AHI decreased from a mean of 8.9 to 2.7
• myofunctional re-education has shown similar reductions in AHI
Longitudinal effect on facial growth after tonsillectomy in children with obstructive sleep apnea. WorldJ.Orthod. 2002; 3, 67–72. Rapid maxillary expansion for pediatric obstructive sleep apnea: A systematic review and meta-analysis. Laryngoscope. 2017 Jul;127(7):1712-1719
What are the barriers for ENTs and Orthodontists collaborating in the management of mouth breathing/ snoring/OSA in Children?
• Lack of understanding of the importance of nasal breathing on facial growth by otolaryngologists• Lack of understanding of the importance of nasal breathing on facial growth by
orthodontists• Lack of established referral patterns between orthodontists and otolaryngologists
• All children should have good nasal breathing prior to starting orthodontics• Not all children with tonsillar hypertrophy need a T&A• Shared decision making between otolaryngologist/ orthodontist/ caregiver is essential
Conclusions
• There may be an association between nasal obstruction and poor facial growth• The research to date is inconclusive• Otolaryngologists should work more closely with orthodontists/ dental
practitioners and consider RME/ myofunctional re-education as options
Case 1:
• 8 yo boy referred by his orthodontist for “large tonsils” and snoring, normal PSG• What do you do? • Shared decision making• Discuss options with orthodontist and family• I would advise T&A
What Do We Do When T&A Fails?
} The options for pediatric OSA post-T&A
} Future trends and research in this field
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Improvements in sleep parameters as measured by PSG
• 80-90% success rate in a meta-analysis of studies up to 2005 • B rietzke SE. The effectiveness of tonsillectom y and adenoidectom y in the treatm ent of pediatric obstructive sleep apnea/hypopnea syndrom e: a m eta-analysis.
O tolaryngol H ead N eck Surg. 2006 Jun;134(6):979-84
• 60-66% success rate in a meta-analysis of studies up to 2009• Friedm an M . U pdated system atic review of tonsillectom y and adenoidectom y for treatm ent of pediatric obstructive sleep apnea/h ypopnea syndrom e.
O tolaryngol H ead N eck Surg. 2009 Jun;140(6):800-8
• Postoperative mean AHI was significantly decreased from preoperative levels in all studies
Causes of Persistent OSA: Normal-weight, no significant comorbidities
• Nasal - the most common cause:
• Allergic Rhinitis
• Adenoid enlargement
• Turbinate hypertrophy
• Septal deviation
Pediatric OSA after T&A: Laryngomalacia
• 2 types of patient populations:
• “classical” supraglottic collapse
• Severe supraglottic collapse
Severe OSA 70%
T&A
No OSA24% (n = 8)
Severe OSA15% (n = 5)
Severe OSA 10% T&A
No OSA80% (n = 28)
Severe OSA0% (n = 0)
Normal-Weight
n=39
Obese n=33
Pediatric OSA after T&A: Obesity
Michelangelo's DAVID goes home to Italy
After a two year visit to the United States , Michelangelo's David is returning to Italy...
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His Proud Sponsors were:OSA and Obesity in Children: What do we do?
• Routine PSG in ALL obese children• Severe OSA (AHI>30; desats <70%)- started on CPAP• T&A within 2 weeks • Severe OSA (AHI>30; desats <70%)- not tolerating CPAP- T&A within 24-48 hours• Nutritional consult/ COACH (Center for Obesity and its Consequences in Health)• PSG at 8-12 weeks • F/U in CPAP clinic (CPAP discontinued if AHI<5 and sats >80%)
Pediatric OSA: beyond T&A in Down Syndrome
Child with DS and OSA
PSG T&A PSG
DISECine MRI
Tongue base Surgery/ Expansion Pharyngoplasty
????Hypoglossal Nerve Stimulator
CPAP
How do we manage a child with DS?Drug Induced Sleep Endoscopy (DISE)
Cine MRI
Novel surgical techniques
• Lingual tonsillectomy
• Base of tongue reduction
• UPPP/ Expansion Palatoplasty
• Hypoglossal nerve stimulators
Who is a candidate for a tongue base procedure?
Children with persistent OSA
• Post T&A• Failed CPAP trial• Endoscopy/ cine MRI shows lingual tonsils/base of tongue
obstruction• Realistic expectations
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Moving the Needle- The Hypoglossal Nerve Stimulator
Maurer et al. Operative technique of upper airway stimulation: an implantable treatment of obstructive sleep apnea. Operative Techniques in Otolaryngology 2012; 23: 227-233.Strollo et al. Upper-airway stimulation for obstructive sleep apnea. NEJM 2014; 270 (2): 139-149.
The Pediatric Hypoglossal Nerve Stimulator
• Initial Pilot Study• Small n (4-6)• Efficacy
• Pilot Study (multicenter)• Slightly larger n (15-20)• Efficacy and outcomes
• Pivotal Study • 50 patients/10 centers
• FDA Approval
Inclusion- Adolescents with Down syndrome age 10 to 21 with prior T&A
- BMI < 95th percentile based on CDC/AAP BMI
growth chart
- Severe OSA: AHI between 10 and 50, and < 25%
central events
- Either have tracheotomy or are unable to tolerate
CPAP
Electrode cuff placed around anterior branches of CN XII
Surgical Pearls
• Staged surgeries are recommended even in moderate to severe OSA
• Using a ‘success’ definition of a postoperative AHI of less than 5, no hypercarbia and no hypoxemia an overall success rate of 50-60% in our small case series of 22 patients
• Predictors of success:• Normal weight• Cine MRI/ DISE shows >
80% obstruction at tongue base
• Should tongue base/palatal procedures be considered in obese children?
• What is the role of hypoglossal nerve stimulators?
QUESTIONS????