Adenopharyngoplasty vs Adenotonsillectomy in Children With Severe Obstructive Sleep Apnea: A Randomized Clinical Trial | Otolaryngology | JAMA Otolaryngology–Head & Neck Surgery | JAMA Network
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Figure 1.  Study Flowchart
Study Flowchart

APP indicates adenopharyngoplasty; ATE, adenotonsillectomy; and OSA, obstructive sleep apnea.

Figure 2.  Adenopharyngoplasty Technique
Adenopharyngoplasty Technique

Closure of the tonsillar pillars after tonsillectomy, with 2 sutures on each side, including fibers of the palatopharyngeus muscle.

Figure 3.  Adenotonsillectomy (ATE) and Adenopharyngoplasty (APP) Group Outcomes
Adenotonsillectomy (ATE) and Adenopharyngoplasty (APP) Group Outcomes

Outcomes of ATE (A) and APP (B). Boxes include the median and the first to third quartile. Whiskers are within the 1.5 interquartile range, and circles are outliers. Line graphs show individual baseline (preoperative) and follow-up (postoperative) obstructive apnea-hypopnea index (OAHI) scores (where a score of 10 or higher is considered severe OSA).

Table 1.  Baseline Characteristics
Baseline Characteristics
Table 2.  PSG Examination and OSA-18 Results From Baseline and the 6-Month Postoperative Follow-up
PSG Examination and OSA-18 Results From Baseline and the 6-Month Postoperative Follow-up
1.
Marcus  CL, Brooks  LJ, Draper  KA,  et al; American Academy of Pediatrics.  Diagnosis and management of childhood obstructive sleep apnea syndrome.  Pediatrics. 2012;130(3):576-584.PubMedGoogle Scholar
2.
Capdevila  OS, Kheirandish-Gozal  L, Dayyat  E, Gozal  D.  Pediatric obstructive sleep apnea: complications, management, and long-term outcomes.  Proc Am Thorac Soc. 2008;5(2):274-282.PubMedGoogle Scholar
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Marcus  CL, Moore  RH, Rosen  CL,  et al; Childhood Adenotonsillectomy Trial (CHAT).  A randomized trial of adenotonsillectomy for childhood sleep apnea.  N Engl J Med. 2013;368(25):2366-2376.PubMedGoogle Scholar
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Mitchell  RB.  Adenotonsillectomy for obstructive sleep apnea in children: outcome evaluated by pre- and postoperative polysomnography.  Laryngoscope. 2007;117(10):1844-1854.PubMedGoogle Scholar
5.
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 Respir Crit Care Med. 2010;182(5):676-683.PubMedGoogle Scholar
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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.PubMedGoogle Scholar
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Friedman  M, Wilson  M, Lin  HC, Chang  HW.  Updated systematic review of tonsillectomy and adenoidectomy for treatment of pediatric obstructive sleep apnea/hypopnea syndrome.  Otolaryngol Head Neck Surg. 2009;140(6):800-808.PubMedGoogle Scholar
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Ye  J, Liu  H, Zhang  GH,  et al.  Outcome of adenotonsillectomy for obstructive sleep apnea syndrome in children.  Ann Otol Rhinol Laryngol. 2010;119(8):506-513.PubMedGoogle Scholar
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Imanguli  M, Ulualp  SO.  Risk factors for residual obstructive sleep apnea after adenotonsillectomy in children.  Laryngoscope. 2016;126(11):2624-2629.PubMedGoogle Scholar
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Costa  DJ, Mitchell  R.  Adenotonsillectomy for obstructive sleep apnea in obese children: a meta-analysis.  Otolaryngol Head Neck Surg. 2009;140(4):455-460.PubMedGoogle Scholar
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Mitchell  RB, Kelly  J.  Outcome of adenotonsillectomy for severe obstructive sleep apnea in children.  Int J Pediatr Otorhinolaryngol. 2004;68(11):1375-1379.PubMedGoogle Scholar
12.
Schellenberg  JB, Maislin  G, Schwab  RJ.  Physical findings and the risk for obstructive sleep apnea. The importance of oropharyngeal structures.  Am J Respir Crit Care Med. 2000;162(2 Pt 1):740-748.PubMedGoogle Scholar
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Schwab  RJ, Gupta  KB, Gefter  WB, Metzger  LJ, Hoffman  EA, Pack  AI.  Upper airway and soft tissue anatomy in normal subjects and patients with sleep-disordered breathing. Significance of the lateral pharyngeal walls.  Am J Respir Crit Care Med. 1995;152(5 Pt 1):1673-1689.PubMedGoogle Scholar
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Guilleminault  C, Li  KK, Khramtsov  A, Pelayo  R, Martinez  S.  Sleep disordered breathing: surgical outcomes in prepubertal children.  Laryngoscope. 2004;114(1):132-137.PubMedGoogle Scholar
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Friedman  M, Samuelson  CG, Hamilton  C,  et al.  Modified adenotonsillectomy to improve cure rates for pediatric obstructive sleep apnea: a randomized controlled trial.  Otolaryngol Head Neck Surg. 2012;147(1):132-138.PubMedGoogle Scholar
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Chiu  PH, Ramar  K, Chen  KC,  et al.  Can pillar suturing promote efficacy of adenotonsillectomy for pediatric OSAS? a prospective randomized controlled trial.  Laryngoscope. 2013;123(10):2573-2577.PubMedGoogle Scholar
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Brodsky  L.  Modern assessment of tonsils and adenoids.  Pediatr Clin North Am. 1989;36(6):1551-1569.PubMedGoogle Scholar
18.
Berry  RB, Budhiraja  R, Gottlieb  DJ,  et al; American Academy of Sleep Medicine; Deliberations of the Sleep Apnea Definitions Task Force of the American Academy of Sleep Medicine.  Rules for scoring respiratory events in sleep: update of the 2007 AASM Manual for the Scoring of Sleep and Associated Events.  J Clin Sleep Med. 2012;8(5):597-619.PubMedGoogle Scholar
19.
Borgström  A, Nerfeldt  P, Friberg  D.  Adenotonsillotomy versus adenotonsillectomy in pediatric obstructive sleep apnea: an RCT.  Pediatrics. 2017;139(4):e20163314.PubMedGoogle Scholar
20.
Borgström  A, Nerfeldt  P, Friberg  D.  Questionnaire OSA-18 has poor validity compared to polysomnography in pediatric obstructive sleep apnea.  Int J Pediatr Otorhinolaryngol. 2013;77(11):1864-1868.PubMedGoogle Scholar
21.
Todd  CA, Bareiss  AK, McCoul  ED, Rodriguez  KH.  Adenotonsillectomy for obstructive sleep apnea and quality of life: systematic review and meta-analysis.  Otolaryngol Head Neck Surg. 2017;157(5):767-773.PubMedGoogle Scholar
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Baldassari  CM, Mitchell  RB, Schubert  C, Rudnick  EF.  Pediatric obstructive sleep apnea and quality of life: a meta-analysis.  Otolaryngol Head Neck Surg. 2008;138(3):265-273.PubMedGoogle Scholar
Original Investigation
July 2018

Adenopharyngoplasty vs Adenotonsillectomy in Children With Severe Obstructive Sleep Apnea: A Randomized Clinical Trial

Author Affiliations
  • 1Department of Otorhinolaryngology, Karolinska University Hospital, Stockholm, Sweden
  • 2Department of Clinical Science, Intervention and Technology, Karolinska Institutet, Stockholm, Sweden
  • 3Department of Otorhinolaryngology, Institute of Surgical Science, Uppsala University, Uppsala, Sweden
JAMA Otolaryngol Head Neck Surg. 2018;144(7):580-586. doi:10.1001/jamaoto.2018.0487
Key Points

Question  Is a modified adenotonsillectomy, termed as adenopharyngoplasty, with closure of the tonsillar pillars more effective than adenotonsillectomy for treating severe obstructive sleep apnea in otherwise healthy children?

Findings  In this randomized clinical trial including 83 children with obstructive sleep apnea who were randomized to receive adenopharyngoplasty or adenotonsillectomy, the mean obstructive apnea-hypopnea index was improved in both groups, and there was no significant difference between the groups. There also were no significant differences between the groups regarding other polysomnography variables or quality of life.

Meaning  Adenopharyngoplasty was not more effective than adenotonsillectomy for treating severe obstructive sleep apnea in otherwise healthy children.

Abstract

Importance  Adenotonsillectomy (ATE) is the primary surgical method for treating obstructive sleep apnea (OSA) in children. However, children with severe OSA have an increased risk for residual OSA after ATE. Previous studies indicate that adenopharyngoplasty (APP), a modified ATE with closure of the tonsillar pillars, might improve the surgical outcome, but the overall evidence is weak.

Objective  To determine whether APP is more effective than ATE for treating severe OSA in otherwise healthy children.

Design, Setting, and Participants  A blinded randomized clinical trial was conducted at the otorhinolaryngology department at Karolinska University Hospital, Stockholm, Sweden. Eighty-three children, aged 2 to 4 years, with an obstructive apnea-hypopnea index (OAHI) score of 10 or higher, were randomized to APP (n = 36) or ATE (n = 47). Participants were recruited from December 1, 2014, through November 31, 2016.

Interventions  Adenotonsillectomy was performed in all 83 patients in both groups by the cold steel technique. The APP group also underwent closure of the tonsillar pillars with 2 inverted sutures on each side.

Main Outcomes and Measures  The primary outcome was the difference between the groups in OAHI score change before and after surgery. A higher score indicates worse problems and a score of 10 or higher is defined as severe OSA. The outcome was evaluated per protocol and with intention-to-treat analysis. Secondary outcomes were other polysomnography variables and the Obstructive Sleep Apnea-18 (OSA-18) questionnaire (possible total symptom score range, 18-126; higher scores indicate worse quality of life). Polysomnography was performed and the OSA-18 questionnaire was completed preoperatively and 6 months postoperatively.

Results  A total of 83 children (49 [59%] boys; mean [SD] age, 36.6 [9.2] months) were included in the study. Of these, 74 (89%) (APP, n = 30; ATE, n = 44) completed the study. The mean (SD) preoperative OAHI score was 23.8 (11.8) for APP and 23.8 (11.5) for ATE. Both the APP and ATE groups had a significant decrease in mean OAHI score after surgery (−21.7; 95% CI, −26.3 to −17.2; and −21.1; 95% CI, −24.5 to −17.7, respectively), but there was no significant difference between the groups (0.7; 95% CI, −4.8 to 6.1). Furthermore, no significant differences between the groups were seen regarding other polysomnography variables (eg, respiratory distress index: mean, 0.6; 95% CI, −5.0 to 6.3) or the OSA-18 questionnaire (eg, total symptom score: −0.5; 95% CI, −13 to 12). One patient from each group was readmitted owing to postoperative bleeding, but no other complications were seen.

Conclusions and Relevance  This trial did not show that APP was more effective than ATE alone to treat otherwise healthy children with severe OSA. This finding suggests that ATE should continue to be the primary treatment for OSA in children.

Trial Registration  ClinicalTrials.gov Identifier: NCT02315911

Introduction

Obstructive sleep apnea (OSA) is a common disorder among children, with prevalence ranging from 1% to 5%, and is associated with numerous morbidities.1 If left untreated, OSA can cause cardiovascular complications, impaired growth, and neurobehavioral disturbances, such as hyperactivity and learning difficulties.1,2 Adenotonsillar hypertrophy is a primary risk factor for OSA, and adenotonsillectomy (ATE) is the first choice of treatment.1,3 Although ATE has a positive effect on respiratory sleep variables, quality of life, and behavior,3 residual OSA is reported to occur in 13% to 75% of the children after surgery.4-8 Risk factors for persistent OSA are severe OSA, obesity, and genetic, neurologic, and craniofacial disorders.4,5,9-11

To improve the surgical outcome, alternative methods have been developed to reduce the obstruction of the upper airway. The soft palate and pharyngeal walls are anatomic factors involved in the obstruction of the upper airway,12,13 and closure of the tonsillar pillars after ATE is believed to enlarge the upper airway. Guilleminault et al14 reported that a modified ATE with closure of the tonsillar pillars resulted in a 100% success rate. In that study and others, the alternative procedure is referred to as adenotonsillectomy with pharyngoplasty14,15 or adenotonsillectomy with tonsillar pillar suturing16; in the present study, it is called adenopharyngoplasty (APP). A randomized clinical trial conducted by Friedman et al15 compared APP (n = 19) with ATE (n = 25). The results of that study were in favor of APP. However, the results from that study showed no significant differences between the groups and the authors reported that the study was underpowered owing to a high dropout rate (27%). Furthermore, a nonrandomized, prospective controlled study by Chiu et al16 showed that APP (n = 12) was significantly more effective than ATE (n = 12), with a reduction in the apnea-hypopnea index (AHI) of 80% for APP compared with 43% for ATE. These studies indicate that APP might be a more effective surgical method than ATE, but the strength of the evidence is weak. The present study aimed to determine whether otherwise healthy children with severe OSA improved more with APP than ATE by analyzing polysomnography (PSG) data and Obstructive Sleep Apnea-18 (OSA-18), a quality-of-life questionnaire, scores.

Methods
Trial Design and Participants

A single-center, blinded, prospective randomized clinical trial with 2 parallel arms comparing ATE with APP was conducted at the Department of Otorhinolaryngology, Karolinska University Hospital in Stockholm, Sweden, with study recruitment from December 1, 2014, through November 31, 2016. Final inclusion was at the day of the operation. All referred children with symptoms of OSA and no characteristics excluding them from study participation were offered a PSG examination. Children who completed the PSG, accepted participation in the study, met the inclusion and exclusion criteria described below, and had written informed consent from their caregivers were candidates for further participation. There was no financial compensation. The protocol is available in the Supplement. This study was approved by the Swedish Regional Ethics Board in Stockholm. The flow of participants is illustrated in Figure 1.

Inclusion and Exclusion Criteria

The following inclusion criteria were used: age, 2 years or older and younger than 5 years; history or symptoms of OSA; severe OSA, defined as an obstructive apnea-hypopnea index (OAHI) score of 10 or higher (no definite maximum level for the OAHI; a higher score indicates worse problems and a score of 10 or higher is defined as severe OSA), tonsil hypertrophy of 2 to 4 according to Brodsky17 (scored according to occlusion of the oropharynx: 1, 0%-25%; 2, 26%-50%; 3, 51%-75%, and 4, 76%-100%); and parents with sufficient knowledge of Swedish to understand the written information and complete the questionnaires. Exclusion criteria were the presence of craniofacial abnormality, neuromuscular disease, chromosomal abnormality, previous adenotonsil surgery, bleeding disorder, and cardiopulmonary disease.

Sample Size and Power Analysis

The sample size was calculated by a statistician prior to the start of the study, with an α level of .05 and 90% power. The power analysis was based on previous studies3,5 showing a reduction of approximately 75% in the OAHI after ATE and a decision that a reduction of 85% or more in the OAHI after APP would be of clinical significance. This percentage generated a study population of 44 children, but this number was increased to 83 children to avoid limitations in the power analysis and compensate for dropouts.

Polysomnography

Polysomnography measures sleep stages and respiratory functions using recordings of electroencephalogram, electrooculogram, electromyogram, pulse, oronasal airflow, transcutaneous oxygen saturation and carbon dioxide, respiratory movements (abdomen and thorax), and body position, as well as video and sound recordings. Polysomnography was performed overnight in a sleep laboratory at the otorhinolaryngology department at baseline and 6 months after surgery. All PSGs were conducted using the same technology (EMBLA; Natus Medical Inc) and scored manually by the same registered polysomnographic technologist according to the scoring rules of the American Academy of Sleep Medicine.18

OSA-18 Questionnaire

The OSA-18 questionnaire is validated to assess the quality of life related to sleep-disordered breathing among children. It consists of 18 questions within the 5 domains of sleep disturbance, physical symptoms, emotional distress, daytime function, and caregivers’ concerns. Each question is scored on a 7-point Likert scale and the responses are summed to a total symptom score ranging from 18 to 126 points, with higher scores indicating a worse quality of life. The OSA-18 also contains a global rating of health-related quality of life on a visual analog scale of 1 to 10 points, with 10 indicating the highest level.

Intervention

The children were randomly assigned to either ATE or APP, and surgery was performed within approximately 3 months after the baseline PSG. Tonsil (according to Brodsky) and adenoid (according to percentage of occlusion of the nasopharynx: 1, 0% to 25%; 2, 26% to 50%; 3, 51% to 75%; and 4, 76% to 100%) size were assessed by the surgeon with the child under general anesthesia at the time of the operation. Tonsillectomy and adenoidectomy were performed in all patients in both groups by the cold steel technique. The tonsils were removed by blunt extracapsular dissection, and the adenoid was removed with a ring knife. In addition, the APP group underwent closure of the tonsillar pillars after the tonsillectomy with 2 inverted sutures (Monocryl 4/0; Ethicon) on each side, including fibers of the palatopharyngeus muscle (Figure 2). A total of 19 surgeons, either specialists or senior residents, were involved.

Hypothesis and Outcomes

The hypothesis of the study is that APP is superior for treating children with severe OSA compared with ATE as measured by the change in OAHI scores 6 months after surgery. The primary outcome was the difference in OAHI score change at the 6-month follow-up between children who underwent ATE and APP.

Secondary outcomes analyzed were differences between the groups regarding changes in other PSG variables, including central apnea-hypopnea index, rapid eye movement apnea-hypopnea index, oxygen desaturation index (using the ≥3% desaturation criteria), and respiratory distress index, which are measured as number of events per hour; mean oxygen saturation, lowest oxygen saturation level, and sleep efficiency, measured as ranges from 0% to 100%; and total sleep time, measured as minutes.

Different levels of postoperative OAHI score (<1, <2, <5, and <10) were analyzed to evaluate the success of surgery. Subgroup analyses were performed regarding children with obesity (body mass index z score ≥1.67 [calculated as weight in kilograms divided by height in meters squared]) and different levels of preoperative OAHI scores (≥20 and ≥30). Regarding the OSA-18 questionnaire, the differences between the groups in changes in total symptom score, sleep disturbance index, and health-related quality of life were analyzed. The need for further surgery because of residual OSA and postoperative complications, such as infection and readmission due to bleeding, was also evaluated.

Randomization and Blinding

The randomization was performed with 120 sealed envelopes stratified into 2 levels: OAHI scores lower than 30 (80 envelopes) and OAHI scores 30 or higher (40 envelopes). The stratification was performed to avoid a skewed distribution of the few children with extreme values (OAHI≥30). The allocation ratio was 1:1 between APP and ATE. Five envelopes from each group were mixed to create a block of 10. The envelopes were placed in the operating room and opened by the surgeon at the day of operation. The surgeon did not meet the child or the caregiver after the operation. The caregivers, patients, researchers, and PSG scorer were blinded to surgical method.

Statistical Analysis

The primary analysis was per protocol, but an intention-to-treat (ITT) analysis was also performed regarding the primary outcome of changes in OAHI score. Missing values were imputed by the standard method in which the last observation was carried forward.

The PSG variables were continuous data, and parametric statistical tests, including paired and unpaired t tests, were used to analyze differences within and between the groups. The results are given as the mean (SD) or as the mean (95% CI). The test of proportion was used when comparing different levels of success.

The OSA-18 questionnaire consisted of ordinal data and was analyzed with nonparametric tests, including the Wilcoxon signed rank test within groups and the Mann-Whitney test between groups. The results are given as the median (interquartile range) or as the median (95% CI). Findings were considered significant at P < .05. All data were analyzed with Stata, version 15 (StataCorp).

Results

A total of 83 children were randomized to either ATE (n = 47) or APP (n = 36) and the groups were similar at baseline Table 1. Mean (SD) age was 36.6 (9.2) months. Four children in the ATE group and 3 children in the APP group were obese (BMI z score ≥1.67). Of the 83 children, 74 (89%) completed the follow-up (ATE group, 44 [94%]; APP group, 30 [83%]).

Primary Outcome

Both groups had a significant reduction in mean OAHI score at the follow-up. The ATE group had a mean decrease of 21.1 (95% CI, 17.7-24.5), the APP group had a mean decrease of 21.7 (95% CI, 17.2-26.3) (decreases of 88% and 91%, respectively), and there was no significant difference between the groups in mean change (0.7; 95% CI, −4.8 to 6.1) (Table 2). All children had a reduction in OAHI, but 1 child in the ATE group still had severe OSA (OAHI score, 27) at the follow-up compared with none in the APP group (Figure 3).

The ITT analysis (n = 83) did not alter the results. The difference between the groups in change in mean OAHI score remained nonsignificant (−1.6, 95% CI, −7.3 to 4.0).

Secondary Outcomes

There were significant improvements within both groups in oxygen desaturation index, rapid eye movement AHI, respiratory distress index, and oxygen saturation nadir, but no significant differences between the groups (Table 2). No significant changes were observed in central AHI level, total sleep time, or sleep efficiency (Table 2).

The success rates at different levels of postoperative OAHI scores (<1, <2, <5, and <10) were 20%, 48%, 84%, and 98%, respectively, in the ATE group compared with 17%, 50%, 90%, and 100%, respectively, in the APP group. There were no significant differences between the groups: 4% (95% CI, −14% to 22%), −2% (95% CI, −25% to 21%), −6% (95% CI, −21% to 9%), and −2% (95% CI, −7% to 2%), respectively. Also, subgroup analyses for children with different preoperative OAHI scores and body mass index z scores showed no significant differences between the groups.

In the OSA-18 questionnaire, significant improvements within both groups were observed in total symptom score, sleep disturbance index, and health-related quality of life (Table 2), but there were no significant differences between the groups. One patient in each group was readmitted owing to postoperative bleeding, but no other complications were seen.

Discussion

Severe OSA is a risk factor for persistent OSA after surgery. This is, to our knowledge, the first randomized clinical trial comparing PSG data between APP and ATE in otherwise healthy children with severe OSA. The results showed that APP was not superior to ATE in this study population regarding change in mean OAHI score, other respiratory PSG variables, and OSA-18 scores. Only 1 of the 47 patients had persistent severe OSA after ATE. This finding suggests that ATE should continue to be the primary treatment for children with severe OSA.

Mean OAHI scores showed a significant reduction in both the APP (91%) and ATE (88%) groups (Table 2), but without a significant difference between the groups. The high treatment effect of 88% in the ATE group could explain the nonsignificant difference because the effect was estimated to be 75% in the power analysis. However, compared with similar studies, the reduction in the ATE group was at a similar level as in a study by Mitchell.4 In that study, 79 children with a mean (SD) preoperative OAHI score of 27.5 (22.5) had a reduction of 87% after ATE. Ye et al8 saw a similar preoperative mean OAHI score of 24.6 (17.3) (n = 84, 89% had an OAHI score ≥10) and had a reduction of 85% after ATE, which further supports the accuracy of the results in the ATE group in the present study.

This study was based on 3 earlier investigations14-16 indicating that APP was more effective than ATE in treating OSA in children, but those studies had some limitations. First, a retrospective study by Guilleminault et al14 reported normal results of PSG examination and no residual symptoms in patients who received APP, but the reported number of participants varied between 4 and 155, which made the results hard to interpret. A prospective study by Chiu et al16 reported a reduction in mean AHI level of 80% after APP compared with 43% after ATE, with a significant difference between the groups. However, the significance of this result could also be questioned because the patients were not randomized, the study sample was small (n = 24), and the preoperative mean AHI values between the groups were not similar (8.3 in the ATE group vs 14.6 in the APP group). Thus, a greater reduction in mean AHI would be expected in the APP group and could explain the significant result between the groups. Finally, the randomized clinical trial by Friedman et al15 that compared APP (n = 19) with ATE (n = 25) showed a postoperative reduction in mean AHI level of 40% in the ATE group compared with 52% in the APP group, but without a significant difference between the groups. Those authors discussed how the lack of significance might be explained by the study being underpowered owing to a high dropout rate (27%). Factors that differed from the present study were that 50% of the patients evaluated by Friedman et al were obese, most did not have severe OSA, only 25% of the sleep studies were scored with PSG, and 75% were scored with peripheral arterial tonometry.

In the present study, there were no significant differences between the groups in success rates given the different postoperative OAHI levels. Friedman et al15 had a success rate of 90% (AHI level <5) after APP in their randomized clinical trial, which was the same as in this study, although their success rate of 72% after ATE was lower compared with the success rate of 84% in the present study. This difference raised the question of whether our results after ATE were unexpectedly high. However, a recent randomized clinical trial by Borgström et al19 between ATE (n = 39) and adenotonsillotomy (n = 40) in a similar group of children with moderate to severe OSA had a similar success rate of 86% (OAHI score <5) after ATE, which is in keeping with the results from the present study.

In the subgroup analyses regarding obesity and different levels of preoperative OAHI scores, there were no significant differences between the groups. However, the present study was not powered to analyze these differences, and further studies of APP vs ATE in children with obesity and a higher mean OAHI score are of interest.

Previous studies have shown that the OSA-18 score does not correlate with the severity of OSA,20 but it can be useful for reporting changes in quality of life after surgery.21,22 In the present study, both groups had significant improvements in OSA-18 scores but no significant differences between the groups (Table 2). The results in the ATE group are consistent with findings in the randomized clinical trial of Borgström et al.19 However, Friedman et al15 also compared OSA-18 scores between APP and ATE and found a significant postoperative difference between the groups in total symptom score in favor of APP. However, this result should be interpreted with caution because there was a significant difference between the groups in mean total symptom score at baseline.

The major strengths of this study are that it was a randomized clinical trial, there was a low dropout rate (11%), it used PSG, and children, caregivers, PSG scorer, and researchers were all blinded to treatment allocation. Also, it is unusual for OSA studies to investigate children between ages 2 and 4 years, even though OSA is most common in this age group.

Limitations

A limitation of this study was the homogeneous group of otherwise healthy children, making the generalizability limited. There were only 7 obese children, and therefore it was not possible to evaluate the effect of APP in obese patients. The randomization process is another limitation that needs to be addressed. There was a skewed distribution between the groups (ATE, n = 47; APP, n = 36). The sealed envelopes were put in a stack, in blocks of 10, and the envelopes were supposed to be taken in order from the top. However, some surgeons might have taken an envelope randomly from the stack, generating a more skewed allocation than expected. Even so, the statistical analysis was not affected, because the children were randomized, the groups were similar at baseline, and there were enough children in each group according to the power analysis.

Conclusions

This randomized clinical trial did not show that APP was more effective than ATE regarding objective PSG variables and OSA-18 scores in this cohort of otherwise healthy children with severe OSA. This finding suggests that ATE should continue to be the primary treatment for severe OSA in children.

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Article Information

Accepted for Publication: March 11, 2018.

Corresponding Author: Johan Fehrm, MD, Department of Otorhinolaryngology B53, Karolinska University Hospital, 141 86 Stockholm, Sweden (johan.fehrm@sll.se).

Published Online: May 31, 2018. doi:10.1001/jamaoto.2018.0487

Author Contributions: Drs Fehrm and Friberg had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.

Study concept and design: Fehrm, Nerfeldt, Friberg.

Acquisition, analysis, or interpretation of data: Fehrm, Sundman, Friberg.

Drafting of the manuscript: Fehrm.

Critical revision of the manuscript for important intellectual content: All authors.

Statistical analysis: Fehrm, Sundman.

Obtained funding: Fehrm, Friberg.

Administrative, technical, or material support: Fehrm, Nerfeldt, Friberg.

Study supervision: Nerfeldt, Friberg.

Conflict of Interest Disclosures: All authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest and none were reported.

Funding/Support: This study was supported by grants from the Stockholm County Council (1211-1594) and the Samaritan Foundation for Pediatric Research.

Role of the Funder/Sponsor: The funding organizations had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.

Additional Contributions: We thank our colleagues at the Department of Otorhinolaryngology, Karolinska University Hospital, for recruiting and operating on the study patients, Anna Borgström, MD, PhD, for providing valuable advice, statistician Johan Bring, PhD, for providing statistical advice, polysomnographic technologist Therese Murphy for interpreting the polysomnographs, and nurse Carina Hedenström and other personnel for assisting in the sleep laboratory. There was no financial compensation outside of salary.

References
1.
Marcus  CL, Brooks  LJ, Draper  KA,  et al; American Academy of Pediatrics.  Diagnosis and management of childhood obstructive sleep apnea syndrome.  Pediatrics. 2012;130(3):576-584.PubMedGoogle Scholar
2.
Capdevila  OS, Kheirandish-Gozal  L, Dayyat  E, Gozal  D.  Pediatric obstructive sleep apnea: complications, management, and long-term outcomes.  Proc Am Thorac Soc. 2008;5(2):274-282.PubMedGoogle Scholar
3.
Marcus  CL, Moore  RH, Rosen  CL,  et al; Childhood Adenotonsillectomy Trial (CHAT).  A randomized trial of adenotonsillectomy for childhood sleep apnea.  N Engl J Med. 2013;368(25):2366-2376.PubMedGoogle Scholar
4.
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