The American Academy of Otolaryngology–Head and Neck Surgery defines sleep-disordered breathing (SDB) as “an abnormal respiratory pattern during sleep, and includes snoring, mouth breathing, and pauses in breathing. SDB encompasses a spectrum of disorders that increase in severity from snoring to obstructive sleep apnea.”1 SDB is associated with symptoms that include excessive sleepiness, inattention, poor concentration, and hyperactivity. Obstructive sleep apnea (OSA) syndrome, as defined by the American Academy of Pediatrics, is a “disorder of breathing during sleep characterized by prolonged partial upper airway obstruction and/or intermittent complete obstruction (obstructive apnea) that disrupts normal ventilation during sleep and normal sleep patterns.”2
The patient was a 12-year-old boy with Down syndrome, OSA, and eustachian tube dysfunction with a 7-year history of SDB. The patient had prior multiple sets of ear tubes and an adenoidectomy for eustachian tube dysfunction. Examination was significant for a tonsil size of 3+ (Table 1). Polysomnography (PSG) showed mild-to-moderate OSA with an apnea/hypopnea index (AHI) of 6.99 events per hour and mixed apnea index of 3.95 events per hour; lowest oxygen saturation was 90%.
Tonsil Size Grading Chart
The patient underwent an intracapsular tonsillectomy with replacement of his ear tubes (minimal, nonobstructive adenoid tissue was noted). Two months postoperatively, his mother noted improved sleep, but a repeat PSG showed residual OSA: an AHI of 2.94 events per hour (down from 6.99), and a rapid eye movement (REM) AHI of 9 events per hour. Oxygen desaturation episodes were mild to moderate and brief, with the exception of one 2-minute-long hypoventilation with saturations in the low 80s. There were periods with end tidal CO2 (ETCO2) around 55 mm Hg, and peak ETCO2 was 57 mm Hg. Continuous positive airway pressure (CPAP) therapy was recommended but was not pursued due to concerns for the patient's inability to tolerate it.
Due to persistent symptoms, the patient underwent placement of long-term ear tubes and a drug-induced sedated endoscopy. Findings included pharyngeal wall cobblestoning, lingual tonsillar hypertrophy, glossoptosis, and a retroflexed epiglottis. The patient was also given topical nasal steroids for nasal obstruction with limited improvement.
Persistent symptoms prompted return to the operating room for an uncomplicated trans-oral endoscopic-assisted coblation lingual tonsillectomy and submucosal reduction/lateralization of his inferior nasal turbinates. At his 2-month follow-up appointment, he had less snoring and fewer night-waking episodes. Residual mild nasal congestion was treated with fluticasone propionate nasal spray.
A repeat PSG 2 years after surgery showed an AHI of 1.6 events per hour (down from 2.94), REM AHI had 3.9 events per hour (previously 9), and no snoring or hypoventilation. The mean oxygen saturation value was 96% and the minimum was 91%. Maximum ETCO2 was 44 mm Hg and average ETCO2 was 41 mm Hg.
The evaluation of children suspected of having SDB starts with a thorough sleep history. Questions posed should include asking about the presence of snoring and its severity, witnessed apneas (including length of pauses witnessed), restless sleep, night time awakenings, nocturia, nasal obstruction, daytime somnolence, hyperactivity, and poor school performance. The questionnaire “I'M SLEEPY” (Table 2) can be a helpful screening tool in the primary care office.3 A medical history should specifically identify any congenital syndromes or illnesses that include low muscle tone (eg, Down syndrome, cerebral palsy). Allergic rhinitis and second-hand smoke exposure should be noted. The physical examination includes a complete head and neck examination to evaluate the upper airway. When tonsil hypertrophy is not noted, a nasopharyngoscopy or laryngoscopy done in the office can help identify specific sites of obstruction.
The “I'M SLEEPY” Questionnaire
The algorithm for the approach to the pediatric patient with suspected OSA differs somewhat among the American Academy of Pediatrics, the American Academy of Sleep Medicine, and the American Academy of Otolaryngology–Head and Neck Surgery.
American Academy of Pediatrics Guidelines
The American Academy of Pediatrics (AAP) clinical practice guidelines2 on the treatment of childhood OSA state that if a child or adolescent snores on a regular basis and has any complaints or findings of gasping, labored breathing, observed apneas, enuresis, cyanosis, daytime sleepiness, attention-deficit/hyperactivity disorder, learning issues, under- or overweight, tonsillar hypertrophy, adenoid facies, micrognathia, high arched palate, failure to thrive, or hypertension, then clinicians should either obtain a PSG or refer the patient to a sleep specialist or otolaryngologist. “If a child with SDB/OSA, has a clinical examination consistent with adenotonsillar hypertrophy, and does not have a contraindication to surgery, the clinician should recommend adenotonsillectomy as the first line of treatment.”2
If the child has OSA but does not have adenotonsillar hypertrophy, other treatments can first be considered, such as CPAP. Additional considerations are recommended for obese children. In a meta-analysis, 10% to 25% of obese children showed a complete resolution of SDB after adenotonsillectomy;4 therefore, CPAP should be considered as a first-line therapy in this population and may be necessary postoperatively if SDB is still present. Weight management is recommended for obese children with OSA pre- and postoperatively. The AAP panel in the guidelines agreed that weight loss is beneficial, “but clinical experience suggests that weight loss is difficult to achieve and maintain, and even effective weight loss regimens take time; therefore, additional treatment is required in the interim.”2
Topical intranasal steroids for patients with persistent OSA postoperatively should also be recommended. They are not recommended as a first-line therapy for OSA due to the incomplete resolution, variability among patients who respond, and lack of long-term data.
American Academy of Sleep Medicine Guidelines
The American Academy of Sleep Medicine states that PSG is indicated in various clinical scenarios.5 PSG is indicated in patients considered for adenotonsillectomy, other upper airway surgeries for OSA, and postoperatively when residual symptoms are present. If the clinical assessment suggests the diagnosis of OSA, congenital central alveolar hypoventilation syndrome or sleep-related hypoventilation due to neuromuscular disorders or chest wall deformities, then PSG should be considered. In addition, positive airway pressure titration, consideration for oral appliance therapy for OSA, and post-treatment evaluations require a PSG. Other scenarios when PSG is indicated include ventilated patients, prior to and after decannulation of a tracheostomy, and in various respiratory disorders (chronic asthma, cystic fibrosis, pulmonary hypertension, bronchopulmonary dysplasia, or chest wall abnormality such as kyphoscoliosis) when there is a clinical suspicion for SDB.5
American Academy of Otolaryngology–Head and Neck Surgery Guidelines
The American Academy of Otolaryngology–Head and Neck Surgery states that PSG is the gold standard for the diagnosis of OSA in the pediatric population.1 PSG is recommended when SDB is suspected in high-risk patients, including those with obesity, Down syndrome, craniofacial abnormalities, neuromuscular disorders, sickle cell disease, and mucopolysaccharidoses. PSG is also recommended if there is uncertainty regarding the diagnosis or if there is smaller than expected adenotonsillar size on examination.1,6 PSG is not needed in otherwise-healthy children (without the above comorbidities) when the diagnosis of SDB is made based on history (which may include audio or video recordings of breathing during sleep) and physical examination.
History suggesting the possibility of SDB includes frequent loud snoring, gasping, snorting, and thrashing in bed, or unexplained bedwetting. Behavioral symptoms can include changes in mood, misbehavior, and poor school performance. Not every child with academic or behavioral issues will have SDB, but if a child snores loudly on a regular basis and is experiencing mood, behavior, or school-performance problems, SDB should be considered.7
Tonsil and adenoid hypertrophy is recognized as the most common cause of SDB in children. Tonsil size is readily identified using a tonsil grading scale (Table 1), with tonsillar hypertrophy defined as grades 3+ or 4+. Combined volume of the tonsils and adenoids correlates with the severity of SDB, although the tonsil size alone does not.8 Tonsils that are only 1+ or 2+ in size may nonetheless contribute to airway obstruction in healthy children, and more so in those with hypotonia or craniofacial anomalies.7
Therapy for Persistent OSA
Adenotonsillectomy is approximately 82% successful as sole management of OSA in children.9 Obesity is the most common risk factor for failure of adenotonsillectomy to cure OSA and resolve SBD in children. In a recent meta-analysis, SDB was completely resolved after adenotonsillectomy in only 10% to 25% of obese children as compared to 70% to 80% of normal-weight children.7 A higher preoperative AHI and older age have also been found to increase the risk of SBD persistence after adenotonsillectomy.10
When a patient shows clinical and/or PSG findings of significant OSA after adenotonsillectomy, CPAP should be recommended as initial treatment for most patients. If the patient cannot tolerate CPAP, additional options for management need to be pursued. For mild OSA/SDB symptoms, management with conservative measures may be worthwhile. For overweight patients, weight-management programming should be included. Allergic and nonallergic rhinitis can be treated with topical nasal steroid sprays and/or oral leukotriene modifiers. Office examination, endoscopy, and/or imaging modalities to assess potential areas of obstruction should be done to help direct management. Oral appliance therapy is a viable option in patients who are able to tolerate the device. Drug-induced sedated endoscopy (DISE) is a valuable tool in investigating sites of persistent obstruction and can aid in surgical decision-making in patients who fail medical management. For example, if soft palate collapse is noted to be the main obstructive issue, then a uvulopalatopharyngoplasty would be a reasonable treatment option.
Multiple other surgical therapies, including a multiple level surgery, may be considered. In the case presented in this article, nasal obstruction due to inferior turbinate hypertrophy and tongue base obstruction secondary to hypertrophic lingual tonsils were identified during the DISE procedure. Aaron et al.11 studied 26 patients (age 3 to 20 years) with persistent OSA after adenotonsillectomy diagnosed with lingual tonsillar hypertrophy. All patients underwent an endoscopic-assisted coblation lingual tonsillectomy. Pre- and postoperative PSG was obtained and analyzed. Statistically significant reductions in the respiratory distress index (RDI) were seen (mean, 14.7 vs 8.1). The RDI is a measurement in the PSG that includes the AHI and respiratory effort-related arousals. There were similar reductions in the number of apneas and hypopneas. The only complications were adhesions between the epiglottis and tongue base, but these appeared to be of no consequence for airway or feeding issues.11 In a systematic review of non-CPAP treatment options for children with persistent pediatric OSA, Manickam et al.12 reviewed 24 articles on the topic. DISE was used much more commonly than cine magnetic resonance to identify sites of obstruction. Lingual tonsillectomy was used alone in six studies (total of 141 patients) with a success rate of 57% to 61% when defined as reducing AHI to <5 events per hour.12
Adenotonsillectomy successfully treats OSA/SDB in the majority of patients, but a subset of pediatric patients will continue to have OSA after adenotonsillectomy. Those with persistent OSA/SDB who remain symptomatic despite medical management and are unable to tolerate CPAP therapy may be considered for additional surgical therapies. DISE can help identify the anatomic cause of residual obstruction and guide additional surgical therapy.
- Roland PS, Rosenfeld RM, Brooks LJ, et al. Clinical practice guideline: polysomnography for sleep-disordered breathing prior to tonsillectomy in children. Otolaryngol Head Neck Surg. 2011;145(1 Suppl):S1–S15. doi:10.1177/0194599811409837 [CrossRef]
- Marcus CL, Brooks LJ, Draper KA, et al. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics. 2012;130(3):576–584. doi:10.1542/peds.2012-1671 [CrossRef]
- Kadmon G, Chung SA, Shapiro CM. I'M SLEEPY: a short pediatric sleep apnea questionnaire. Int J Pediatr Otorhinolaryngol. 2014;78(12):2116–2120. doi:10.1016/j.ijporl.2014.09.018 [CrossRef]
- Costa DJ, Mitchell R. Adenotonsillectomy for obstructive sleep apnea in obese children: a meta-analysis. Otolaryngol Head Neck Surg. 2009;140(4):455–460. doi:10.1016/j.otohns.2008.12.038 [CrossRef]
- Wise MS, Nichols CD, Grigg-Damberger M, et al. Executive summary of respiratory indications for polysomnography in children: an evidence-based review. Sleep. 2011;34(3):389–398.
- Alexander NS, Schroeder JW. Pediatric obstructive sleep apnea syndrome. Pediatr Clin North Am. 2013;60(4):827–840. doi:10.1016/j.pcl.2013.04.009 [CrossRef]
- Baugh RF, Archer SM, Mitchell RB, et al. Clinical practice guideline: tonsillectomy in children. Otolaryngol Head Neck Surg. 2011;144(1 suppl):S1–30. doi:10.1177/0194599810389949 [CrossRef]
- Arens R, McDonough JM, Costarino AT, et al. Magnetic resonance imaging of the upper airway structure of children with obstructive sleep apnea syndrome. Am J Respir Crit Care Med. 2001;164(4):698–703. doi:10.1164/ajrccm.164.4.2101127 [CrossRef]
- Brietzke SE, Gallagher D. The effectiveness of tonsillectomy and adenoidectomy in the treatment of pediatric obstructive sleep apnea/hypopnea syndrome: a meta-analysis. Otolaryngol Head Neck Surg. 2006;134(6):979–984. doi:10.1016/j.otohns.2006.02.033 [CrossRef]
- Tauman R, Gulliver TE, Krishna J, et al. Persistence of obstructive sleep apnea syndrome in children after adenotonsillectomy. J Pediatr. 2006;149(6):803–808. doi:10.1016/j.jpeds.2006.08.067 [CrossRef]
- Aaron C, Lin AC, Koltai PJ. Persistent pediatric obstructive sleep apnea and lingual tonsillectomy. Otolaryngol Head Neck Surg. 2009;141(1):81–85. doi:10.1016/j.otohns.2009.03.011 [CrossRef]
- Manickam PV, Shott SR, Boss EF, et al. Systematic review of site of obstruction identification and non-CPAP treatment options for children with persistent pediatric obstructive sleep apnea. Laryngoscope. 2016;126(2):491–500. doi:10.1002/lary.25459 [CrossRef]
- Friedman M, Ibrahim H, Bass L. Clinical staging for sleep-disordered breathing. Otolaryngology Head Neck Surg. 2002;127(1):13–21. doi:10.1067/mhn.2002.126477 [CrossRef]
Tonsil Size Grading Chart
|1+: tonsils are hidden within the pillars
|2+: tonsils are extending to the pillars
|3+: tonsils are extended beyond the pillars but not to the midline
|4+: tonsils extend to the midline
The “I'M SLEEPY” Questionnaire
I – Is your child frequently irritated or angry during the day?
M – Is the child's body mass index >85%?
S – Does your child usually snore?
L – Does your child sometimes have labored breathing at night?
E – Ever noticed a stop in your child's breathing during sleep?
E – Does your child have enlarged tonsils and/or adenoids?
P – Does your child have problems with concentration?
Y – Does your child often yawn or seem sleepy during the day?
I – Are you angry a lot?
M – Filled in by the doctor: body mass index above 85%?
S – Do you snore at night?
L – Did your parents or a friend tell you that your breathing is “difficult” at night?
E – Did your parents or a friend tell you that you stop breathing at night?
E – Do you have problems with your tonsils or adenoids (glands inside your mouth)?
P – Is it difficult for you to focus (at school or at home)?
Y – Do you feel tired or sleepy a lot?