BEYOND T&A: TREATMENT OF RESIDUAL SLEEP APNEA IN CHILDREN Cecille G. Sulman, MD Associate Professor Chief, Division of Pediatric Otolaryngology

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Risk factors for recurrence of OSA • • • • • •

Severe pre-operative OSA Gain velocity in BMI Obesity African American Age > 7 Asthma

Amin Am. J. Respir. Crit. Care Med. 2008 ;177 (6):654-659 Costa DJ, Mitchell R. Adenotonsillectomy for obstructive sleep apnea in children: a meta-analysis. Otol-Head & Neck Surgery (2009)140(4)455-60. Bhattacharjee R, Kheirandish-Gozal L, Spruyt K, et al. Adenotonsillectomy outcomes in treatment of obstructive sleep apnea in children: a multicenter retrospective study. Am J Resp Crit Care Med (2010)182:676-83. Mitchell RB, Kelly J. Adenotonsillectomy for obstructive sleep apnea in obese children. Otolaryngol Head Neck Surg (2004)131:104-108.

Indications for post-op sleep study • Age < 3 • Cardiac complications of OSA • Craniofacial anomalies • Obesity

• • • •

Severe pre-operative PSG Failure to thrive Neuromuscular disorders Persistent sleep disordered breathing

American Academy of Pediatrics. Clinical practice guideline: diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics (2002)109(4)704-712.

12 yo obese male with Down syndrome. • Snoring at night, worsening over past 3 years. • Adenotonsillectomy at 7 years of age. • PSG AHI 56.

S1. How would you further evaluate this patient? A. No further evaluation required; recommend CPAP B. Sleep endoscopy and surgical intervention C. Imaging – plain film, cine MRI D. Both sleep endoscopy and imaging E. Other

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Sleep endoscopy • Supine position without a shoulder roll, mimicking the position of natural sleep as much as possible. • Induction with inhalational mask anesthesia. • Dexmedetomidine infusion at 1-2 mcg/kg/hr, concurrent ketamine bolus of 1 mg/kg . • Less muscular relaxation and a more sustained respiratory effort with this current technique compared to propofol. • Oxymetazoline:1% xylocaine on a 1 cm × 4 cm cottonoid pledget. • Spontaneous respiration supported by nasal cannula oxygen. Lin and Koltai. Sleep endoscopy in the evaluation of pediatric obstructive sleep apnea. International Jour of Ped 2012. doi:10.1155/2012/576719.

Sleep endoscopy

No apnea

Circumferential collapse

Sleep endoscopy • Adult outcomes: – Valid, reliable method to evaluate site, degree, and configuration of upper airway obstruction – Findings are associated with outcomes of palate surgery (Level of evidence 4). • Children outcomes: – Oropharynx/lateral walls - most common site of obstruction with single site obstruction. – Combined oropharynx/lateral walls and velum obstruction - most common sites of obstruction with multiple site obstruction. – Sleep endoscopy directed surgery improves OSA (limited evidence). Rodriguez-Bruno K, Goldberg AN, McCulloch CE, Kezirian EJ. Test-retest reliability of drug-induced sleep endoscopy. Otolaryngol Head Neck Surg 2009;140:646–651. Kezirian EJ, White DP, Malhotra A, Ma W, McCulloch CE, Goldberg AN. Interrater reliability of drug-induced sleep endoscopy. Arch Otolaryngol Head Neck Surg 2010;136:393–397. Iwanaga K, Hasegawa K, Shibata N, et al. Endoscopic examination of obstructive sleep apnea syndrome patients during drug-induced sleep. Acta Otolaryngol Suppl 2003:36–40.

Imaging • High kilovoltage lateral neck imaging – adenoid and tonsil hypertrophy. • Chest radiography - evidence of pulmonary hypertension or right ventricular hypertrophy in a child with severe obstructive sleep apnea. • Cine magnetic resonance imaging - observe airway collapse in 3 planes (axial, coronal and sagittal) to isolate anatomic sites of airway obstruction in children who have persistent apnea after T&A. Donnelly LF, Shott SR, LaCrosse CR, et al. Causes of persistent obstructive sleep apnea despite previous tonsillectomy and adenoidectomy in children with Down syndrome as depicted on static and dynamic cine MRI. Am J Roentgenol (2004)183(1):175-81. Shott SR, Donnelly LF. Cine magnetic resonance imaging evaluation of persistent airway obstruction after tonsil and adenoidectomy in children with Down syndrome. Laryngoscope (2004)114:1724-9.

S2. What surgical interventions have you performed for OSA after T&A? A. Lingual tonsillectomy only B. Palatal procedures only C. Tongue base procedures – midline glossectomy, repose, geniohyoid suspension only D. Two or more of these techniques

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Sleep Surgery Options in Children • Nasal cavity

– Septoplasty – Turbinate reduction

• Palate

– Pillarplasty – Expansion sphincter pharyngoplasty – Uvulopalatopharyngoplasty

• Tongue – – – –

Glossectomy Lingual tonsillectomy Radiofrequency ablation Genioglossus stabilization (Repose)

• Craniofacial – – – –

Genioglossus advancement Geniohyoid suspension Mandibular advancement Rapid maxillary expansion

• Airway

– Supraglottoplasty – Tracheostomy

Turbinate Volume Reduction • Techniques: Radiofrequency ablation and microdebrider-assisted reduction. • Both techniques for nasal obstruction are effective, however maintenance of improvement at 2 years better with microdebriderassisted technique (Level 1 evidence). • Post-operative course: Mild-to-moderate edema with subsequent nasal obstruction and thick mucus formation for about a week. • Complications: If mucosal erosion is present, the risk of postoperative bleeding and adherent crust formation increases with radiofrequency ablation. Leong SC, Kubba H, White PS. A review of outcomes following inferior turbinate reduction surgery in children for chronic nasal obstruction. Int J Pediatr Otorhinolaryngol (2010)74(1):1-6. Liu CM, Tan CD, Lee FP, Lin KN, et al. Microdebrider-assistedversus radiofrequency-assisted inferior turbinoplasty. Laryngoscope (2009)119(2):414-418. Kezirian EJ, Powell NB, Riley RW, et al. Incidence of complications in radiofrequency treatment of the upper airway. Laryngoscope (2005)115(7):1298-304.

Pillarplasty • Oversew anterior and posterior tonsillar pillars over the tonsillar fossa after tonsillectomy. • Reduces the collapsibility of the pharynx • Limited evidence: Comparison of children with Down syndrome, the addition of pillarplasty (lateral pharyngoplasty) did not improve outcomes. Merrell J, Shott S. OSAS in Down syndrome: T&A versus T&A plus lateral pharyngoplasty International Journal of Pediatric Otorhinolaryngology (2007) 71, 1197—1203 http://www.sleepapneasurgery.com/images/PPP-intra-op-lg.jpg

8 year old male with snoring • Adenotonsillectomy age 3, never stopped snoring • Difficulty paying attention in school, always fatigued • AHI 8 • Mallampati 4; sleep endoscopy with elongated palate no lingual tonsil hypertrophy or tongue base collapse

S3. What options would you consider? A. CPAP B. UPPP C. Expansion sphincter pharyngoplasty 0% A.

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Expansion Sphincter Pharyngoplasty • Rotation and suspension of the palatopharyngeus muscle onto the soft palate, sparing the uvula. • Stabilizes palate and improves diameter of the oral airway. • May be incorporated into the initial surgical approach with tonsillectomy or as a secondary procedure in patients who have persistent sleep apnea after T&A. • Outcomes: reduction in AHI in adults; evidence not available for children. Guilleminault C, Li K, Quo S, et al. A prospective study on the surgical outcomes of children with sleepdisordered breathing. Sleep (2004)27:95-100. Pang KP, Woodson BT. Expansion sphincter pharygnoplasty: a new technique for the treatment of obstructive sleep apnea. Otolaryngol Head Neck Surg (2007)137(1):110-4.

Expansion sphincter pharyngoplasty

Woodson Operative Techniques Otol 2012;23:3–10

Uvulopalatopharyngoplasty • Removal of the soft palate and uvula, widens oropharynx • Limited evidence in children: – Reported success in children with cerebral palsy and hypotonic upper airway muscles. – Lack of substantial reports in normal children.

• Complications: nasopharyngeal stenosis, palatal incompetence, and speech difficulties. Sied AB, Martin PJ, Pransky SM, et al. Surgical therapy of obstructive sleep apnea in children with severe mental insufficiency. Laryngoscope (1990)100(5):507-10. Abdu MH, Feghali JG. Uvulopalatopharyngoplasty in a child with obstructive sleep apnea. J Laryngol Otol (1988)102:5465-8. Carenfelt C, Haradsson PO. Frequency of complications after uvulopalatopharyngoplasty. Lancet (1993)341:437.

Glossectomy • Decrease tongue volume and proportionally increase airway size. • Population: Beckwith-Wiedemann or Down syndrome. • Techniques: open or via a submucosal minimally invasive technique • Success rates for the submucosal technique - 60% • Complications: airway edema, hematoma, abscess formation, and permanent hypoglossal injury Wooten CT, Shott SR. Evolving therapies to treat retroglossal and base-of-tongue obstruction in pediatric obstructive sleep apnea. Arch Otolaryngol Head Neck Surg (2010)136:983-7. Conacher, D, Meikle D, O'Brien C. Trachesotomy, lingual tonsillectomy and sleep-related breathing disorders. Br J Anesth 2002;88(5):724-6.

Midline glossectomy

Woodson Operative Techniques Otol 2012; 23:2:155–161

13-1/2-year-old with Down syndrome • Adenotonsillectomy age 8. • BMI 24.9 (94%). • AHI 11.8

S4. What surgical intervention(s) would you recommend? A. B. C. D. E.

CPAP Lingual tonsillectomy Repose Genioglossus advancement Combination therapy 0% A.

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Lingual Tonsillectomy

Repose Procedure

Outcomes of Multilevel Surgery • Little research is available regarding outcomes of multilevel surgeries in children • 8.2% incidence of oropharyngeal scarring and stenosis in 48 children who underwent multilevel surgery that included lingual tonsillectomy for OSA in children. • Solitary surgical improvements in airway size can augment airway dynamics and reduce the Bernoulli and Starling effects and collapse at other levels. • A staged approach should be considered in children Prager Arch Otolaryngol Head Neck Surg. November 15, 2010,136(11):1111-1115 Marcus Pediatric Research (2005) 57, 99–107

14 yo female with Down syndrome • T&A, lingual tonsillectomy UPPP age 4 • Preop AHI 27, Postop AHI 1.2 • Morbid obesity • Unable to tolerate bipap 21/17

Intraoperative Findings • Oropharyngeal stenosis, 1 cm opening • Normal bronchoscopy

Oropharyngeal Stenosis

Anesthesia Concerns with Severe OSA • During induction: high risk for airway obstruction, desaturation and laryngospasm. • Abnormal ventilator response to carbon dioxide • Greater respiratory depression in response to sedatives, narcotics, and general anesthetics. • Delay in the return to spontaneous ventilation and emergence from general anesthesia • Presence of trace volatile anesthetics will further reduce what may be preexisting abnormal ventilatory drive and potentiate airway obstruction due to reduced function of the genioglossus and other airway muscles. • Risk for post-extubation obstruction, laryngospasm, desaturation, pulmonary edema, and respiratory arrest. Knill RL, Clement JL. Site of selective action of halothane on the peripheral chemoreflex pathway in humans. Anesthesiology (1984)61:121-6. Sanders JC, King MA. Perioperative complications of adenotonsillectomy in children with obstructive sleep apnea. Anesthesia & Analgesia (2006)103(5):1115-21. McColley SA, April MM, Carroll JL, Loughlin GM. Respiratory compromise after adenotonsillectomy in children with obstructive sleep apnea. Arch Otolaryngol Head Neck Surg (1992)118:940-3.

Discussion • Multiple options exist beyond T&A • Procedure selection guided by physical examination and sleep endoscopy • Post-op sleep studies and follow up – Long term follow up for at-risk patients • Delayed recurrence • Stenosis

– Patients who report symptom resolution may still have OSA – Help understand characteristics of patients that may determine surgical success or failure

Thank you

Supplemental slides

Snoring in children • Snoring occurs in 3 – 27% of children.

– Primary snoring has implications with associated morbidity of elevated blood pressure and reduced arterial distensibility. – Obstructive sleep apnea (OSA) occurs in 40% of children who snore.

• Prevalence increasing over time with increased prevalence of obesity. Schecher MS, Section on Pediatric Pulmonology, Subcommittee on Obstructive Sleep Apnea Syndrome. Technical report: diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics (2002)109:e69. Owen GO, Canter RJ, Robinson A. Snoring, apnoea, and ENT symptoms in the paedriatric community. Clin Otolaryngol (1996)21:130-4. O'Brien LM, Sitha S, Bau LA, Waters KA. Obesity increases the risk for persisting obstructive sleep apnea after treatment in children. Int J Pediatr Otorhinolaryngol (2006)70:1555-60.

Why does OSA matter? • Associated morbidities: – – – – – – –

Pulmonary hypertension Cor pulmonale Failure to thrive Growth retardation Behavioral disturbances Poor school performance Enuresis

Brouillette et al. Obstructive sleep apnea in infants and children. Pediatrics 1982;100(1):31-40.

Adenotonsillectomy outcomes • Success rates for T&A range from 59.8% to 100%, with a significant improvement in AHI from preoperative levels. • Overall improvement in quality of life, academic performance. • Postoperative reports of symptoms such as snoring and witnessed apneas correlate well with persistence of OSA after T&A. Clinical Practice Guidelines: Management Pediatric OSA Pediatrics 2004;109(4):704-712. Lipton AL, Gozal D. Treatment of obstructive sleep apnea in children: do we really know how? Sleep Med Rev (2003)7:61-80. Tran et al. Sleep and quality of life in children. Arch Otol Head Neck Surg 2005;131:52-57. De Serres LM, Derkay C, Sie K, et al. Impact of adenotonsillectomy on quality of life in children with obstructive sleep disorders. Otolaryngol Head Neck Surg (2002)128:48996. Suen JS, Arnold JE, Brooks LJ. Adenotonsillectomy for treatment of obstructive sleep apnea in children. Arch Otolaryngol Head Neck Surg (1995)121:525-30 Costa DJ, Mitchell R. Adenotonsillectomy for obstructive sleep apnea in children: a meta-analysis. Otol Head & Neck Surgery (2009)140(4)455-60.

Adenotonsillectomy outcomes

Childhood Adenotonsillectomy (CHAT) Study • Multisite prospective randomized controlled trial. • 464 children with PSG documented mild-moderate SDB were randomized to either T&A or watchful waiting for 7 months. • AHI normalized in 30 – 50% in the control group over seven months without surgical intervention. • Subjects with more severe obstructive sleep apnea showed a larger treatment effect. • T&A was associated with a more significant improvement in measures of sleep quality and sleep disruption, such as arousal index or hypercapnia.

Adenotonsillectomy outcomes

Childhood Adenotonsillectomy (CHAT) Study • T&A group improved over controls in: – Quality of life measures for obstructive symptoms – Behavioral measures (regulation, academics, and internalizing behavior).

• The primary end point, the attention executive function domain of the NEPSY, did not show a statistically significant difference between the two groups. Redline S; Amin R; Beebe D; Chervin RD; Garetz SL; Giordani B; Marcus CL; Moore RH; Rosen CL; Arens R; Gozal D; Katz ES; Mitchell RB; Muzumdar H; Taylor HG; Thomas N; Ellenberg S. The Childhood Adenotonsillectomy Trial (CHAT): rationale, design, and challenges of a randomized controlled trial evaluating a standard surgical procedure in a pediatric population. SLEEP 2011;34(11):1509-1517.

Septoplasty • Careful patient selection (> age 6 years) with limited approach. • Useful in improving CPAP tolerance, particularly in older children. • Complications: persistent septal deviation, bleeding, and septal perforation. • Pediatric septoplasty can be performed without affecting most aspects of nasal and facial growth. Not performing or delaying septoplasty when indicated may adversely affect nasal and facial growth with compounding adverse effects in terms of deformity and asymmetry. Lawrence R. Pediatric septoplasty: a review of the literature. Int J Pediatr Otorhinolaryngol. 2012 Aug;76(8):1078-81.

Genioglossus advancement • Midline osteotomy of the mandible, advance and secure the tongue with plates.

– Not amenable in small children due to presence of tooth buds.

• Repose: pass suture through the tongue base, stabilize to the medial aspect of the mandible with a screw. • Complications: wound infection, edema, and seromas. • Long term effectiveness unknown at this time. Riley R, Guilleminault C, Powell N, et al. Mandibular osteotomy and hyoid bone advancement for obstructive sleep apnea: a case report. Sleep (1984)7(1):79-82. DeRowe A, Gunther E, Fibbi A, et al. Tongue-base suspension with a soft tissue-to-bone anchor for obstructive sleep apnea: preliminary clinical results of a new minimally invasive technique. Otolaryngol Head Neck Surg (2000)122:100-3. Powell NB, Riley RW, Guilleminault C. Radiofrequency tongue base reduction in sleep=disordered breathing: a pilot study. Otolaryngol Head Neck Surg (1999)120(5):656-64.

Geniohyoid suspension

Radiofrequency ablation • Insertion of a 2-pronged probe, generates thermal damage at multiple points in the tongue base. • Tongue bulk and flaccidity of the tongue base is reduced through fibrosis. • Complications (3.4%): range from mucosal ulceration, to superficial infection, and transient parasthesia of the hypoglossal nerve. Farrar J, Ryan J, Oliver E et al. Radiofrequency ablation for the treatment of obstructive sleep apnea: a meta-analysis. Laryngoscope (2008)118(10):1878-83.

Mandibular distraction • In combination with adenotonsillectomy, maxillary distraction has a cure rate of 87.5% in children with sleep apnea. • May avert the need for tracheostomy in children with Pierre Robin sequence, less successful in children with complex congenital syndromes. • Complications: premature callus consolidation, cheek abscess requiring incision and drainage, minor lip erosion from pin contact, facial cellulitis, unilateral facial paralysis, and temporal mandibular joint ankylosis.

Bouchard C, Troulis MJ, Kaban LB. Management of obstructive sleep apnea: role of distraction osteogenesis. Oral Maxillofacial Surgery Clinics of North America (2009)21(4):459-75. Ow AT, Cheung LK. Meta-analysis of mandibular distraction osteogenesis: clinical applications and funtional outcomes. Plast Reconstru Surg (2008)121(3):54e-69e. Mandell DL, Yellow RF, Bradley JP, et al. Mandibular distraction for micrognathia and severe upper airway obstruction. Arch Otolaryngol Head Neck Surg (2004)130(3):344-8.

Mandibular distraction

Rapid maxillary expansion • Oral appliance adjusted daily to increase palatal width. • High-arched palates with associated – Increased nasal resistance – Posterior tongue posture – Retroglossal airway narrowing and mild OSA

• Most effective in pre-pubertal children prior to palatal suture closure. • May be used in combination with adenotonsillectomy to improve the nasal and oral airway Cistulli PA, Palmisano RG, Poole MD. Treatment of obstructive sleep apnea syndrome by rapid maxiallary expansion. Sleep (1998)21:831-5. Wang X, Wang XX, Liang C, et al. Distraction osteogenesis in correction of micrognathia accompanying obstructive sleep apnea syndrome. Plast Reconstr Surg (2003)112:1549-57. Guilleminault C, Quo S, Huynh NT, et al. Orthodontic expansion treatment and adenotonsillectomy in the treatment of obstructive sleep apnea in prepubertal children. Sleep; (2008)31:953-7.

Laryngomalacia • Primarily seen in infancy, but may present in older children • Consider supraglottoplasty in the setting of OSA, failure to thrive, or feeding difficulties • Medical comorbidities are associated with worsened postoperative outcomes, although the majority of children improve after supraglottoplasty Sulman CS, Holinger LD. Stridor, Aspiration and Cough. In: Bailey BJ and Johnson JT, editors. Head and Neck Surgery – Otolaryngology, 4th ed. Philadelphia: Lippincott Williams & Wilkins (2006) p. 1095-118. Chen DK, Truong MT, Koltai PJ. Supraglottoplasty for occult laryngomalacia to improve obstrucive sleep apnea syndrome. Archives of Otolaryngology Head Neck Surg (2012)138(1):50-54.

Laryngomalacia