Summary Laryngomalacia is the most common cause of stridor in neonates and infants. Associated feeding difficulties are present in approximately half of the children. A definitive diagnosis can ...generally be made with flexible fiberoptic laryngoscopy. The disorder is most often self-limited with resolution of symptoms within the first 24 months of life, and the majority of children can thus be managed conservatively. The approximately 5%-20% of children with severe or refractory disease may require more aggressive intervention, most commonly in the form of trans-oral supraglottoplasty 1,2 . High success rates and a low rate of complications have been reported for this procedure in otherwise healthy children. Children with syndromes or medical comorbidities are more likely to have complications or persistent symptoms after supraglottoplasty and may require additional interventions.
Background Adenotonsillectomy (AT) is commonly performed for childhood OSA syndrome (OSAS), but little is known about prognosis without treatment. Methods The Childhood Adenotonsillectomy Trial ...(CHAT) randomized 50% of eligible children with OSAS to a control arm (watchful waiting), with 7-month follow-up symptom inventories, physical examinations, and polysomnography. Polysomnographic and symptomatic resolution were defined respectively by an apnea/hypopnea index (AHI) <2 and obstructive apnea index (OAI) <1 and by an OSAS symptom score (Pediatric Sleep Questionnaire PSQ) < 0.33 with ≥ 25% improvement from baseline. Results After 194 children aged 5 to 9 years underwent 7 months of watchful waiting, 82 (42%) no longer met polysomnographic criteria for OSAS. Baseline predictors of resolution included lower AHI, better oxygen saturation, smaller waist circumference or percentile, higher-positioned soft palate, smaller neck circumference, and non-black race (each P < .05). Among these, the independent predictors were lower AHI and waist circumference percentile < 90%. Among 167 children with baseline PSQ scores ≥ 0.33, only 25 (15%) experienced symptomatic resolution. Baseline predictors were low PSQ and PSQ snoring subscale scores; absence of habitual snoring, loud snoring, observed apneas, or a household smoker; higher quality of life; fewer attention-deficit/hyperactivity disorder symptoms; and female sex. Only lower PSQ and snoring scores were independent predictors. Conclusions Many candidates for AT no longer have OSAS on polysomnography after 7 months of watchful waiting, whereas meaningful improvement in symptoms is not common. In practice, a baseline low AHI and normal waist circumference, or low PSQ and snoring score, may help identify an opportunity to avoid AT. Trial Registry ClinicalTrials.gov; No.: NCT00560859; URL: www.clinicaltrials.gov.
Each year, over 500,000 adenotonsillectomies (AT), mostly for the treatment of pediatric obstructive sleep apnea (OSA) are performed in the US in children under 15 years of age. No definitive study, ...however, has been yet conducted that has rigorously evaluated the effectiveness of AT for not only improving sleep disordered breathing, but also for improving clinically relevant outcomes, such as neurocognitive function, behavior, and quality of life. The Childhood Adenotonsillectomy Trial (CHAT) was designed to assess neuropsychological and health outcomes in children randomized to receive early AT (eAT) as compared to Watchful Waiting with Supportive Care (WWSC). Important secondary goals of the study are to evaluate outcomes in subgroups defined by obesity and race. This paper addresses key elements in the design and implementation of a controlled trial for a widely used "standard practice" surgical intervention in a pediatric population, that include establishment of standardized data collection procedures across sites for a wide variety of data types, establishment of equipoise, and approaches for minimizing unblinding of selected key personnel. The study framework that was established should provide a useful template for other pediatric controlled studies or other studies that evaluate surgical interventions.
To summarize published studies that evaluate whether adenotonsillectomy performed for sleep-disordered breathing in children is associated with improvements in behavior, cognitive function, and ...quality of life, whether those improvements show correlation with polysomnographic parameters, and suggest how future studies may provide additional clinically significant information.
A computerized search of the medical literature was performed for articles published between 1950 and March 2007 with the use of the OVID Medsearch database.
Analysis revealed 25 articles that satisfied the inclusion and exclusion criteria. All studies showed improvement in one or more of the specified outcome measures including general or disease specific quality of life, behavioral problems including hyperactivity and increased aggression or neurocognitive skills, such as memory, attention, or school performance. Limited correlation was often seen between improvements in outcome measures and polysomnographic variables.
Current studies strongly suggest adenotonsillectomy performed for sleep-disordered breathing in children is associated with improvements in quality of life, behavior, and cognitive function, but large, randomized, controlled studies are needed to provide definitive evidence of the benefits of this commonly performed surgical procedure in the general population.
Data from a randomized, controlled study of adenotonsillectomy for obstructive sleep apnea syndrome (OSAS) were used to test the hypothesis that children undergoing surgery had greater quality of ...life (QoL) and symptom improvement than control subjects. The objectives were to compare changes in validated QoL and symptom measurements among children randomized to undergo adenotonsillectomy or watchful waiting; to determine whether race, weight, or baseline OSAS severity influenced changes in QoL and symptoms; and to evaluate associations between changes in QoL or symptoms and OSAS severity.
Children aged 5 to 9.9 years with OSAS (N = 453) were randomly assigned to undergo adenotonsillectomy or watchful waiting with supportive care. Polysomnography, the Pediatric Quality of Life inventory, the Sleep-Related Breathing Scale of the Pediatric Sleep Questionnaire, the 18-item Obstructive Sleep Apnea QoL instrument, and the modified Epworth Sleepiness Scale were completed at baseline and 7 months. Changes in the QoL and symptom surveys were compared between arms. Effect modification according to race and obesity and associations between changes in polysomnographic measures and QoL or symptoms were examined.
Greater improvements in most QoL and symptom severity measurements were observed in children randomized to undergo adenotonsillectomy, including the parent-completed Pediatric Quality of Life inventory (effect size ES: 0.37), the 18-item Obstructive Sleep Apnea QoL instrument (ES: -0.93), the modified Epworth Sleepiness Scale score (ES: -0.42), and the Sleep-Related Breathing Scale of the Pediatric Sleep Questionnaire (ES: -1.35). Effect modification was not observed by obesity or baseline severity but was noted for race in some symptom measures. Improvements in OSAS severity explained only a small portion of the observed changes.
Adenotonsillectomy compared with watchful waiting resulted in significantly more improvements in parent-rated generic and OSAS-specific QoL measures and OSAS symptoms.
Polysomnography defines the pathophysiology of obstructive sleep apnea syndrome (OSAS) but does not predict some important comorbidities or their response to adenotonsillectomy. We assessed whether ...OSAS symptoms, as reflected on the Sleep-Related Breathing Disorders Scale of the Pediatric Sleep Questionnaire (PSQ), may offer clinical predictive value.
Baseline and 7-month follow-up data were analyzed from 185 participants (aged 5-9 years with polysomnographically confirmed OSAS) in the surgical treatment arm of the multicenter Childhood Adenotonsillectomy Trial. Associations were assessed between baseline PSQ or polysomnographic data and baseline morbidity (executive dysfunction, behavior, quality of life, sleepiness) or postsurgical improvement.
At baseline, each 1-SD increase in baseline PSQ score was associated with an adjusted odds ratio that was ∼3 to 4 times higher for behavioral morbidity, 2 times higher for reduced global quality of life, 6 times higher for reduced disease-specific quality of life, and 2 times higher for sleepiness. Higher baseline PSQ scores (greater symptom burden) also predicted postsurgical improvement in parent ratings of executive functioning, behavior, quality of life, and sleepiness. In contrast, baseline polysomnographic data did not independently predict these morbidities or their postsurgical improvement. Neither PSQ nor polysomnographic data were associated with objectively assessed executive dysfunction or improvement at follow-up.
PSQ symptom items, in contrast to polysomnographic results, reflect subjective measures of OSAS-related impairment of behavior, quality of life, and sleepiness and predict their improvement after adenotonsillectomy. Although objective polysomnography is needed to diagnose OSAS, the symptoms obtained during an office visit can offer adjunctive insight into important comorbidities and likely surgical responses.
Abstract
Study Objectives
Obstructive sleep apnea is associated with neurobehavioral dysfunction, but the relationship between disease severity as measured by the apnea-hypopnea index and ...neurobehavioral morbidity is unclear. The objective of our study is to compare the neurobehavioral morbidity of mild sleep-disordered breathing versus obstructive sleep apnea.
Methods
Children 3–12 years old recruited for mild sleep-disordered breathing (snoring with obstructive apnea-hypopnea index < 3) into the Pediatric Adenotonsillectomy Trial for Snoring were compared to children 5–9 years old recruited for obstructive sleep apnea (obstructive apnea-hypopnea 2–30) into the Childhood Adenotonsillectomy Trial. Baseline demographic, polysomnographic, and neurobehavioral outcomes were compared using univariable and multivariable analysis.
Results
The sample included 453 participants with obstructive sleep apnea (median obstructive apnea-hypopnea index 5.7) and 459 participants with mild sleep-disordered breathing (median obstructive apnea-hypopnea index 0.5). By polysomnography, participants with obstructive sleep apnea had poorer sleep efficiency and more arousals. Children with mild sleep-disordered breathing had more abnormal executive function scores (adjusted odds ratio 1.96, 95% CI 1.30–2.94) compared to children with obstructive sleep apnea. There were also elevated Conners scores for inattention (adjusted odds ratio 3.16, CI 1.98–5.02) and hyperactivity (adjusted odds ratio 2.82, CI 1.83–4.34) in children recruited for mild sleep-disordered breathing.
Conclusions
Abnormal executive function, inattention, and hyperactivity were more common in symptomatic children recruited into a trial for mild sleep-disordered breathing compared to children recruited into a trial for obstructive sleep apnea. Young, snoring children with only minimally elevated apnea-hypopnea levels may still be at risk for deficits in executive function and attention.
Trial Registration
Pediatric Adenotonsillectomy for Snoring (PATS), NCT02562040; Childhood Adenotonsillectomy Trial (CHAT), NCT00560859
Research reveals mixed evidence for the effects of adenotonsillectomy (AT) on cognitive tests in children with obstructive sleep apnea syndrome (OSAS). The primary aim of the study was to investigate ...effects of AT on cognitive test scores in the randomized Childhood Adenotonsillectomy Trial.
Children ages 5 to 9 years with OSAS without prolonged oxyhemoglobin desaturation were randomly assigned to watchful waiting with supportive care (n = 227) or early AT (eAT, n = 226). Neuropsychological tests were administered before the intervention and 7 months after the intervention. Mixed model analysis compared the groups on changes in test scores across follow-up, and regression analysis examined associations of these changes in the eAT group with changes in sleep measures.
Mean test scores were within the average range for both groups. Scores improved significantly (P < .05) more across follow-up for the eAT group than for the watchful waiting group. These differences were found only on measures of nonverbal reasoning, fine motor skills, and selective attention and had small effects sizes (Cohen's d, 0.20-0.24). As additional evidence for AT-related effects on scores, gains in test scores for the eAT group were associated with improvements in sleep measures.
Small and selective effects of AT were observed on cognitive tests in children with OSAS without prolonged desaturation. Relative to evidence from Childhood Adenotonsillectomy Trial for larger effects of surgery on sleep, behavior, and quality of life, AT may have limited benefits in reversing any cognitive effects of OSAS, or these benefits may require more extended follow-up to become manifest.
Obstructive sleep apnea syndrome (OSAS) has been associated with cardiometabolic disease in adults. In children, this association is unclear. We evaluated the effect of early adenotonsillectomy (eAT) ...for treatment of OSAS on blood pressure, heart rate, lipids, glucose, insulin, and C-reactive protein. We also analyzed whether these parameters at baseline and changes at follow-up correlated with polysomnographic indices.
Data collected at baseline and 7-mo follow-up were analyzed from a randomized controlled trial, the Childhood Adenotonsillectomy Trial (CHAT).
Clinical referral setting from multiple centers.
There were 464 children, ages 5 to 9.9 y with OSAS without severe hypoxemia.
Randomization to eAT or Watchful Waiting with Supportive Care (WWSC).
There was no significant change of cardiometabolic parameters over the 7-mo interval in the eAT group compared to WWSC group. However, overnight heart rate was incrementally higher in association with baseline OSAS severity (average heart rate increase of 3 beats per minute bpm for apnea-hypopnea index AHI of 2 versus 10; standard error = 0.60). Each 5-unit improvement in AHI and 5 mmHg improvement in peak end-tidal CO2 were estimated to reduce heart rate by 1 and 1.5 bpm, respectively. An increase in N3 sleep also was associated with small reductions in systolic blood pressure percentile.
There is little variation in standard cardiometabolic parameters in children with obstructive sleep apnea syndrome (OSAS) but without severe hypoxemia at baseline or after intervention. Of all measures, overnight heart rate emerged as the most sensitive parameter of pediatric OSAS severity.
Clinicaltrials.gov (#NCT00560859).
PDF
