Ped e iat a r t i r c c S lee e p e A p A nea e a Surg r er e y

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Airway Imaging in Pediatric OSA Kasey Li, MD, DDS, FACS Stanford University Sleep Medicine Program The airway is smaller in children with OSA compared to controls The adenoid and tonsils are larger and the airway is most restricted where adenoid and tonsils overlap The volume of adenotonsillar tissues correlates with AHI Arens R, et al. Am J Respir CCM 2003;167:65-70. 70. Arens R, et al. Am J Respir CCM 2001;164:698-703. Tonsillectomy/Adenoidectomy Surgical Outcomes The cure rate of T&A for OSA is 80% - 401 patients cumulative data from 11 studies RDI > 19 was more likely to have Post-op op RDI > 5 Shen et al. Arch Oto H N Surg 1995;44:51. Obese children, positive family history of OSA and African American Children are at high risk for having residual OSA after T&A Morton S, et al. Sleep 2001;24:823. 1

Surgical Outcomes 110 children with OSA underwent T&A 25% children achieve AHI 1, 46% AHI <5, 29% had AHI 5 Obesity and AHI are the major determinant for outcome Tauman R, et al. J Pediatr 2006;149:803-808. 808. Singulair and CPAP are treatment options for children with persistent OSA following T&A Interview of Dr. David Gozal Recurrence 23 pubertal & postpubertal teenagers w/ prior T&A 5 subjects (all boys) reported snoring, 3 with OSA Mandibular deficiency/diminished posterior airway seen with the OSA patients Guilleminault C, et al. J Pediatrics 114;997, 1989. 20 subjects followed 12 years after T&A (20 controls) Increased snoring in the T&A group (50% vs. 20%) Greater inspiratory effort in the T&A group in PSG Diminished airway space persists 12 years after T&A Tasker C, et al. Arch Dis Child 86;34, 2002. CPAP Retrospective study - 79 Children (6 mo-18 yr) 65 (82%) accepted CPAP, (34 took 9-295 days) 6-12 yr > 13-18 18 yr > below 6 yr Mean follow up 207 days (8-979 days) 50 (mean age 10 yr) had objective data (counter) 78% had complicating medical disorder 76% used CPAP at least half the days ( 1 hr) Mean daily use 4.7 hrs 5 yo 7.2 hrs, 6-12 yo 4.2 hrs, 13-1818 yo 3.6 hrs O Donnell AR, et al. Sleep 29;651-658, 658, 2006. CPAP Retrospective study - 46 Children (7-19 yr) Mean age 13.6, mean BMI 39.8 (29 obese, 6 Down) 27/46 (59%) pts had objective data Mean follow up 18.1 mo (3-43 mo) 19/27 (70%) compliant (adult criteria) Parents over estimated usage 4 hrs 2 hrs > 4 hrs 0.5 hrs Uong EC, et al. Pediatrics 120;e1203-11, 11, 2007. 2

CPAP Prospective study - 29 children (2-16 yr) at 3 centers Randomly assigned CPAP/BiPAP 8 (28%) dropped out in 6 mos 21 with objective/subjective data Mean nightly use 5.3 ± 2.5 hrs Parents over estimated use (7.6 hrs vs 5.8 hrs) 78% of parents admitted that CPAP not used nightly Subjective improvement in daytime sleepiness High drop out rate/suboptimal use Marcus CL, et al. Pediatrics 117;e442-51, 2006. & Nasal Obstruction Nasal obstruction is a risk factor for SDB in children Urschitz, et al. Chest 126:790, 2004. Corbo GM, et al. Pediatrics 108:1149, 2001. Anuntaseree W, et al. Pediatr Pulmonl 32:222, 2001. Treatment of nasal obstruction improves SDB Mansfield LE, et al. Ann Allergy 92:240, 2004. Nixon GM, et al. Am J Respir Med 1:159, 2002. Nasal obstruction leads to increased nasal resistance and mouth breathing, which can negatively affect facial growth McNamara JA. Angle Orthod 50:269, 1981. Harvold EP, et al. Am J Orthod 79:359, 1981. Turbinate Reduction by RF 10 patients (15 mm electrode w/ 2 lesions) Improvement: 75%, (L) 68% No prescription pain meds needed 8/9 patients w/ preop meds were discontinued Laryngoscope 109:683, 1999. 24 patients (15 mm electrode w/ 2 lesions) Improvement: severity 86%, frequency 94% No adverse effect on nasal physiology Evaluation: VAS, acoustic rhinometry, rhinomanometry, butanol threshold test, sacchrine test, olfaction, ciliary beat frequency Laryngoscope 111:153, 2001. 14 patients (10 mm electrode w/ 3 lesions) Improvement (VAS): obstructin (7-3) Evaluation: acoustic rhinometry, sacchrine, ciliary test Laryngoscope 111:894,2001. Stanford Sleep Disorders Clinic experience 2004-20062006 Adenotonsillectomy/Pharyngoplasty Pharyngoplasty/Turbinate reduction N=189, mean age 9.8 (1-1717 years) Tonsils: 9-1+, 97-2+, 76-3+, 7-4+ All patients underwent T&A/pharyngoplasty 86 patients underwent RF turbinate reduction One patient with minor bleeding 10 days postop secondary to severe coughing-no no tx 330 (87.3%) tonsillar wounds remained completely closed 3

Stanford Sleep Disorders Clinic experience 2004-20062006 Improvement of turbinate size 2.98 ± 0.99 to 0.91 ± 0.76 2 minor crusting Bleeding rate - 0.5% Control of bleeding 0% Dehydration 0% Readmission 0% Airway Involvement 26 snoring children studied by PSG and 26 controls 96.2% of OSA children are mouth breathers Vertical facial growth pattern identified in OSA group Retroposition and postinclination of the mandible 69% w/ labial incompetence and 52% w/ crossbite in OSA group, none in controls Diminished nasopharyngeal airway (enlarged adenoids) in OSA group Zucconi M, et al. Eur Respir J 13;411, 1999. OSA children have craniofacial abnormalities (increase ant. lower facial height, retrognathic mandible ) Kawashina S, et al. Acta Paediatr 91;71, 2002. 4

Maxillary Morphology 40 OSA patients and 21 controls Dental casts, cephalometrics evaluated 50% of OSA patients vs. 5% of controls with posterior transverse discrepancies OSA patients have narrower, more tapered and shorter maxillary arch Seto BH, et al. Eur J Orthod 23;703; 2001. Maxillary Morphology 13 patients with Marfan s syndrome (high arch palate) and 13 controls Maxilla significantly narrower (ICD, IPD, IMD) in Marfan s group Nasal resistance abnormal in 12 Marfan s and 4 controls Mean nasal resistance was doubled in Marfan s group 7 in Marfan s group with OSA, none in controls Cistulli PA, et al. Chest 110:1184, 1996. Nasal Airway & Rapid Maxillary Expansion Retrospective review of 10 children (6.5-15.5 y.o ) with h/o nocturnal enuresis who underwent RME All patients with nasal obstruction NE improved, nasal breathing (subjective) improved in all patients Timms DJ. Angle Orthod 60:229, 1990. Prospective study of 10 children (8-13 y.o) with NE Six patients are mouth breathers, 8 Class II, 1 crossbite NE improved in 7 patients, nasal resistance improved in all Kurol J, et al. Angle Orthod 68:225, 1998. Nasal Airway & Rapid Maxillary Expansion Retrospective review of 237 patients 91% improvement (subjective) of nasal breathing Timms DJ. L Laryngol Otol 98:357, 1984. Prospective study of 26 patients (age 10-20) with RME Nasal resistance evaluated by rhinomanometry Amount of expansion 5-9 mm (1 st molar) Reduction of resistance in all (mean 36% improvement) Weak correlation between improvement and expansion Timms DJ. Br J Orthod 13;221, 1986. 5

Rapid Maxillary Expansion Prospective Study w/ 10 OSA patients (range 19-43 y.o) All have maxillary constriction w/ dental crowding 6 patients w/ surgically assisted RME Mean expansion 12.1 mm at the appliance 9/10 patients improved (RDI 19 to 7, LSAT 89% to 91%) Cistulli PA, et al. Sleep 21:831, 1998. Rapid Maxillary Expansion Prospective Study of 31 children with OSA 19 boys, mean age 8.7 years, mean AHI 12.2 (5.7-21.1yo) Maxillary constriction, no adenotonsillar hypertrophy 22 had prior T & A 9 Class I, 14 Class II and 8 Class III RME- mean expansion 4.32 mm All had increased nasal resistance (26 bi-, 5 unilateral) Decrease in nasal resistance in all children AHI < 1 in all children Pirelli, Saponjara, Guilleminault. Sleep 27:761, 2004. Orthopedic effect of RME w/ maxillary processes opening in a triangular fashion-base at the palatal vault Am J Orthod Dentofac Orthop 107:268, 1995. 6

Orthodontic Expansion 16 successively treated children mean age 7.9 years, with a combination of enlarged tonsils (grade 2+ and 3+) and clinically narrow maxilla were involved in the study. All children were seen by Pediatric sleep specialist, Otolaryngologist, Oral/Maxillofacial surgeon and Orthodontist Pre R.M.E 2.6mm Post R.M.E 8.3mm 7

Results Variables Pre Post Mean AHI 12.2 ±4.0 5.5 ±3.7 Mean RDI 21 ± 4.9 7 ± 5.1 Lowest SaO 2 92 ± 2.3 96 ± 2.2 TST (min.) 421 ± 14 418 ±17 Before After 8

Surgery Airway and Maxillomandibular Expansion 9

Patien t MME by Distraction Osteogenesis for OSA Guilleminault and Li. Laryngoscope 114:893, 2004 Sex Age Pre RDI Post RDI Pre Pes Post Pes Pre O2 Post O2 Size (mm) F/U (mo) 1 F 24 4.3 0-13 -5 92 96 7 11 2 M 18 2.2 1.6-14 -7 89 93 12 25 3 M 9 9 0.7 - - 88 91 7 30 4 M 18 0.8 1.9-33 -12 90 90 12 25 5 6 M F 21 43 Mean 22 21.4 41.2 8.4 15 - - - - 84 86 86 92 12 7 13 5 18 22 13.2 4.5-20 -8 88 91 9.5 18 Maxillomandibular Advancement Adult MMA Data 50/50 (100%) success rate Mean age 42.7 years Mean BMI changed from 30.7 to 28.6 Mean RDI improved from 59.2 to 4.7 Mean LSAT improved from 72.7% to 88.6 Chest 1999; 116:1519-1529. 1529. 15/20 (75%) success rate Mean age 45 years Mean BMI changed from 26.9 to 25.4 Mean RDI improved from 59 to 11 Mean LSAT improved from 82% to 90% Am J Respir Crit Care Med 2000;162:641-649. 649. 10

Adult MMA Results Retrospective review 320 patients 306 patients (95%) with postop PSG 272 patients had preoperative RDI > 30 Age: 45.0 ± 9.5 years BMI: 32.7 ± 6.7 kg/m 2 RDI: 63.6 ± 26.7 to 10.5 ± 10.0 LSAT: 71.5 ± 15.1% to 86.9 ± 6.7% Success rate: 273 patients (89%) Adult MMA Results 33 patients (11%) are incomplete responder Age: 45.0 ± 7.3 years BMI: 35.0 ± 7.2 kg/m 2 RDI: 64.1 ± 25.2 to 32.2 ± 11.4 LSAT: 70.7 ± 16.5% to 81.5 ± 6.7% 32 patients in severe category pre-op 17 patients in severe category post-opop Adult MMA Results 502 MMA results evaluated Retrospective data 306 patients Prospective data 196 patients No mortality 88% success rate RDI < 20 and/or at Least a Reduction in RDI of 50% SaO2 > 90% or a Minimum Fall Below 90% Quality of life improvement 9 children (7 boys) persistent OSA All non-obese, obese, non-syndromic Mean age 14.7 ± 2 (10-1717 yr) Mean RDI 26.7 ± 17.5 (9-65.6) Mean LSAT 89.8 ± 1.2% (88-91%) CPAP recommended as initial tx 3 refused CPAP and 6 failed CPAP 7 MMA, 2 mandibular advancement 11

Variables Pre-Tx Post-Tx RDI 26.7 ± 17.5 3.9 ± 3.1 Lowest SaO 2 89.8 ± 1.2 91.8 ± 1.3 No complications encountered Daytime fatigue and sleepiness improved in all patients but two patients continue to have residual symptoms Follow up 26.7 ± 17.5 months Case Presentation #1 16 year old boy Long history of daytime fatigue. Does not remember when he is not tired. Behavior problems and lives with grandparents Currently undergoing orthodontia Adenotonsillectomy at age 6 PSG RDI 65.6 events per hour LSAT 90% Case Presentation 12

Case Presentation #2 10 year old boy Long history of daytime fatigue. Behavior, social and school problems PSG RDI 22 events per hour LSAT 89% Struggling with PAP for the past 4 years 13

Case Presentation #3 15 year old girl with significant daytime fatigue and tiredness for years. 7 years of orthodontic therapy PSG RDI 62 events per hour LSAT 84% CPAP recommended and initiated Mother/patient elected to proceed with MMA ASAP after 4 months of CPAP 14

Surgical Outcomes Case #1: PSG: RDI-1, LSAT 94% Improvement of quality of sleep as well daytime fatigue Case #2: PSG: RDI-2, LSAT 92% Improvement of behavior, social and school issues Case #3: PSG: RDI-6, LSAT 92% Resolution of all symptoms Conclusions Significant number of pediatric patients have continual problems and symptoms following Adenotonsillectomy Many children are unable to tolerate CPAP Most children are suboptimally treated with CPAP therapy Improvement of nasal airway by RF turbinate reduction, skeletal expansion by orthodontic or surgery provide additional treatment options for patients beyond adenotonsillectomy 15