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Effect of drug induced sleep endoscopy on intraoperative decision making in pediatric sleep surgery
Journal Pre-proof Effect of Drug Induced Sleep Endoscopy on Intraoperative Decision Making in Pediatric Sleep Surgery Julia Dmowska, Stephen Reed Larson, M. Boyd Gillespie, Anthony Sheyn PII:
S0165-5876(19)30563-4
DOI:
https://doi.org/10.1016/j.ijporl.2019.109810
Reference:
PEDOT 109810
To appear in:
International Journal of Pediatric Otorhinolaryngology
Received Date: 3 September 2019 Revised Date:
29 November 2019
Accepted Date: 30 November 2019
Please cite this article as: J. Dmowska, S.R. Larson, M.B. Gillespie, A. Sheyn, Effect of Drug Induced Sleep Endoscopy on Intraoperative Decision Making in Pediatric Sleep Surgery International Journal of Pediatric Otorhinolaryngology, https://doi.org/10.1016/j.ijporl.2019.109810. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Elsevier B.V. All rights reserved.
Effect of Drug Induced Sleep Endoscopy on Intraoperative Decision Making in Pediatric Sleep Surgery
Author names and affiliations Julia Dmowska1 Stephen Reed Larson1 M. Boyd Gillespie1 Anthony Sheyn1 1. Department of Otolaryngology, The University of Tennessee Health Science Center, 910 Madison Ave. Memphis, TN 38163; USA Corresponding author Julia Dmowska1 1. Department of Otolaryngology, The University of Tennessee Health Science Center, 910 Madison Ave. Memphis, TN 38163; USA. [email protected].
Conflicts of interest The authors report no conflicts of interest.
Abstract Objectives: To demonstrate the effect of drug induced sleep endoscopy (DISE) on intra-operative decision making during pediatric sleep surgery for obstructive sleep apnea (OSA). Methods: A retrospective chart review was performed on pediatric (3-17 years) patients with moderate-to-severe OSA (7.2-71.8) who underwent drug induced sleep endoscopy at the time of initial sleep surgery. The characteristics evaluated included age, race, gender, site of obstruction, type of surgical intervention, preand post-operative apnea and hypopnea index. Of the 26 patients that were identified, 18 had both a preand post-operative polysomnograms results. Results: All patients underwent DISE immediately prior to surgical treatment. The mean pre-operative AHI for the 18 patients with post-operative polysomnogram results was 21.3 (7.2-71.8). The mean post-operative AHI for the 18 patients was 7.6 (0.7-25.1). There was a significant difference between pre- and post-operative AHI (p < 0.001 ). Of the 26 patients, the most common area of collapse was the soft palate, occurring in 17/26 (65.4%) patients. Base of tongue involvement was found to be present in 11/26 (42.3%) patients, and the epiglottis was involved in 4/26 (15.4%). Evidence of multilevel collapse was observed in 6/26 (23.1%) patients. Patients observed to have palatal collapse underwent a pharyngoplasty (20/26; 76.9%) at the time of adenotonsillectomy. Three (11.5%) patients underwent a tongue reduction.
Conclusion: This study provides additional evidence that DISE can affect intra-operative decision making, with the potential for improved post-operative outcomes. A randomized controlled study is needed to determine if these outcomes are better than what can be achieved without DISE. Keywords Drug induced sleep endoscopy Obstructive sleep apnea Pediatric sleep apnea Adenotonsillectomy
Subdivisions 1. Introduction 2. Materials and methods 3. Results 4. Discussion 5. Conclusions
1. Introduction Obstructive sleep apnea (OSA) is one of the most commonly diagnosed pediatric sleep disorders [1]. Prevalence of this disorder is between 1-3% but may be as high as 5-6% [2]. OSA is associated with repeated episodes of partial or complete airway obstruction during sleep that leads to blood gas abnormalities and sleep fragmentation [1]. This disorder is most commonly associated with adenotonsillar hypertrophy in children [1]. During childhood, the adenoids and tonsils enlarge at a faster rate than the bony structures of the nasopharynx resulting in a narrowed upper airway with increased airflow resistance and recurring upper airway collapse [1]. Adenotonsillar hypertrophy, however, is not always associated with OSA [1]. Additionally, OSA may also be due to other anatomical structures including nasoseptal obstruction, enlarged soft palate or uvula, macroglossia, hypotonic pharynx, micrognathia, maxillary hypoplasia, and lingual tonsil hypertrophy [3]. The current first line treatment, when there is physical evidence of adenotonsillar hypertrophy, is adenotonsillectomy [3]. However, up to 15 to 20% of children have persistent OSA after primary adenotonsillectomy [2, 4, 5]. Persistence of OSA may be due to obesity and/or multifactorial obstruction of the upper airway [5, 6, 7]. Additional surgical options for persistent OSA after adenotonsillectomy are dependent on the site of obstruction and include pharyngoplasty, uvulopalatopharyngoplasty, base of tongue resection, lingual tonsillectomy, septoplasty and supraglottoplasty [6]. Therefore, the ability to identify the site(s) of obstruction prior to an initial surgery would seem to be advantageous to both the patient and surgeon. Drug induced sleep endoscopy (DISE) was introduced in 1991 to provide direct visualization of upper airway collapse under anesthesia [8]. It has been shown to be a safe and useful tool to determine the site, pattern, and degree of upper airway obstruction [5, 9]. Findings include the severity of obstruction in the palatal and hypopharyngeal regions, the collapse of structures including the tongue, epiglottis, velum, and lateral walls, as well as the effect of mouth opening on the airway lumen [7]. In adults, studies have demonstrated that DISE influences choice of therapy and may lead to increased surgical success for the
initial treatment of OSA [8, 10, 11]. DISE may be considered a standard procedure to perform on adults with severe OSA. It has a significant impact on treatment recommendations in adults and guides toward therapeutic options other than CPAP [11]. Informed by the findings on DISE, the surgeon can initiate site specific surgical therapy to treat complex OSA [5]. In pediatrics, DISE is currently used for patients who are non-responsive to initial surgery for OSA [4, 7]. It is largely reserved as a second line diagnostic tool to determine why adenotonsillectomy was not completely effective. One major criticism of DISE in the pediatric population is that it requires additional anesthesia time outside of standard operative management [5]. The current study investigates the utility of DISE during primary sleep surgery for pediatric OSA. The rationale was that DISE would permit observation of upper airway obstruction intra-operatively thereby guiding the initial surgical management. The hope was to achieve improved patient outcomes as defined by post-operative polysomnography with less need for repeated anesthesia. 2. Materials and methods IRB approval was obtained from the University of Tennessee Health Science Center and a retrospective chart review was performed on patients from Le Bonheur Children’s Hospital. Eligible subjects were under 18 years of age at the time of evaluation; met criteria for moderate-severe OSA on a pre-operative polysomnogram with an apnea-hypopnea index (AHI) ≥ 5 and underwent DISE at the time of primary sleep surgery. It was standard practice to consent the care giver of the patient that underwent DISE to include all procedures as “possible.” During the consent process, all families underwent a discussion about the risks and complications of each procedure that could possibly be performed during the time of surgery depending on the findings of the drug induced sleep endoscopy. These procedures included: drug induced sleep endoscopy, adenotonsillectomy, pharyngoplasty, lingual tonsillectomy and base of tongue reduction. Patient and disease related variables were abstracted from the electronic medical records included age, race, gender, BMI, site of obstruction, type of surgical intervention and preand post-operative AHI. During pre-operative examination only adenotonsillar hypertrophy was noted on
physical exam; thus, physical exam findings were not included in the variables reported. Duration of DISE was not specifically measured; the timing was included in the total surgical time. DISE findings were classified at the level of obstruction. Additional surgeries that were performed after DISE depending on findings included: adenotonsillectomy; expansion pharyngoplasty consisting of a tonsillar pillar suture using a vertical mattress suture to connect the posterior tonsillar pillar to the soft palate; and lingual tonsillectomy with midline base of tongue reduction utilizing coblation. This was accomplished by using a Benjamin laryngoscope to expose the base of tongue and using coblation on a setting of 9/3. A pairedsamples t-test was conducted to compare pre-operative and post-operative AHI in patients that had postoperative polysomnogram follow-up. 3. Results A total of 26 patients were identified with documented DISE findings that met the inclusion criteria. The characteristics of the patients were evaluated. The age of the patients ranged from 3 to 17 years old with a mean age of 8.76 (s.d.±4.35). The gender distribution included 8 (30.8%) females and 18 (69.2%) males. Racial background was 22 (84.6%) African Americans, 3 (11.5%) Hispanics and 1 (3.8%) white. Mean BMI was 25.4 (13.9-46.2) with equivalent BMI for age percentile of 75.6% (females 81.9%, males 77.3%). A total of 16/26 (55.1%) patients had medical comorbidities. Six patients had heart conditions (arrhythmia, PFO), 6 had endocrine abnormalities (acanthosis nigricans, hyperinsulinemia, T2DM), 3 had upper airway irregularities (asthma), and 3 had a learning delay (developmental, speech) (Table 1). Sleep endoscopy findings were reviewed (Table 2). The most common area of collapse was the soft palate, occurring in 17 out of 26 patients (65.4%). The base of the tongue was involved in 11 patients (42.3%), with findings of hypertrophy, collapse and obstruction. Retroflexion of the epiglottis was found in 4 patients (15.4%). Tonsillar hypertrophy was found in 19 patients (73.1%). Multilevel collapse was present in 6 out of the 26 patients (23.1%).
Operations were performed for 26 patients (Table 2). All but one patient underwent an adenotonsillectomy (96.2%). Twenty patients (76.9%) underwent a pharyngoplasty at the time of the adenotonsillectomy. Two patients underwent a simultaneous tongue reduction with adenotonsillectomy due to base of tongue hypertrophy and obstruction. One additional patient underwent a tongue reduction operation without adenotonsillectomy. Twenty-six patients had a pre-operative polysomnogram. The average pre-operative AHI was 22.1 (s.d. ±14.0). Post-operative polysomnograms were obtained 3-6 months post-op. A total of 18 patients had post-operative polysomnogram. The average pre-operative AHI, for the patients that had a postoperative polysomnogram results, was 21.3 (sd ±14.3). The average post-operative polysomnogram AHI for the 18 patients was 7.6 (s.d. ±7.4) (Table 3). A paired-samples t-test was conducted to compare preoperative and post-operative AHI of these 18 patients. There was a significant difference in AHI scores (t(17)= 5.05, p < 0.001 ) indicating improvement of AHI after DISE-directed surgery. Eight patients did not have a post-operative polysomnogram. Twenty patients had clinical follow up. Two of the patients with clinical follow up declined a postoperative polysomnogram. At clinical follow up, 12/20 (60.0%) patients had complete resolution of their symptoms according to caregiver confirmation and 5/20 (25.0%) patients had improvement with mild residual symptoms. Two patients (10.0%) had persistent symptoms and one patient (5.9%) had worsening symptomatology which included increased snoring and apneic pauses. No patients required follow-up surgeries. A total of 6/26 (23.1%) patients were lost to follow-up. No adverse events or complications due to the DISE or multi-level airway surgery were reported. No complications occurred due to additional procedures to adenotonsillectomy. Similarly, additional anesthesia time did not adversely affect the patients. 4. Discussion
The patients in this series had many of the recognized risk factors for OSA. Known risk factors include obesity, craniofacial abnormalities, neuromuscular dysfunction, prematurity, Down syndrome, achondroplasia, cerebral palsy and myelomeningocele [1]. Additionally, African American children are more likely than white children to have both OSA and OSA of a more severe degree [1]. Of the charts that were reviewed, 85% of the patients were African American. The most common comorbidities included were cardiac and endocrine in nature. OSA can lead to pulmonary hypertension which can worsen heart disease. Therefore, if heart comorbidities are present, further investigation with DISE into the anatomical cause of the OSA could allow early initiation of treatment to prevent worsening of comorbid conditions. OSA can impair quality of life for children. It may lead to symptoms such as snoring, cessation of breathing, cyanosis and enuresis with consequences of daytime somnolence, behavioral changes, poor academic outcomes, hyperactivity, decline in memory performance and pulmonary hypertension [1, 2, 3, 12]. Early treatment implementation increases the possibility of reversing the complications of OSA [1]. The patients reviewed had OSA symptoms of snoring, daytime somnolence, and to a lesser degree, speech delay. All of these characteristics are consistent with the description of OSA. A total of 17 patients had a BMI percentile greater than 95%. Hence, a majority (65%) of the patients reviewed were obese. Obesity is the most significant risk factor for OSA [1, 13]. Childhood obesity can increase risk for OSA by more than fourfold and OSA can be present in up to 50% of obese children [13, 14]. Obesity is also associated with higher apnea and hypopnea indexes [13]. The American Academy of Pediatrics recommends adenotonsillectomy as the first line treatment for OSA in obese children [14]. Nevertheless, residual OSA will occur in 54-76% of these children [14]. Persistent OSA is more common in obese children compared to normal weight children [15]. Obesity is a profound risk factor for OSA and is associated with residual symptoms after the recommended first line treatment. Based on the characteristics of a patient, especially BMI, it is possible to determine the type of patient that would warrant further surgery, in addition to adenotonsillectomy, to resolve OSA symptoms. These patients
often have multifactorial obstruction and it would be advantageous to use DISE to identify additional sites of obstruction. In this study, patients undergoing DISE were found to have a variety of obstructions of the upper airway. Soft palate collapse, tongue collapse and involvement of the epiglottis were the most common findings outside of adenotonsillar hypertrophy. The current consensus is to perform adenotonsillectomy as the initial operative treatment for OSA [3]. The childhood adenotonsillectomy trial (CHAT) evaluated the efficacy of adenotonsillectomy versus watchful waiting [16]. The trial concluded that there was a greater proportion of normalization of polysomnographic findings in the patients that underwent surgery [16]. Additionally, patients with more severe cases of OSA had larger absolute improvements in polysomnographic findings with surgical intervention [16]. However, there is a recognition that patients with severe OSA are less likely to show normalization compared to less severe cases and that residual disease is very common [5, 6, 7, 16, 17]. The mean AHI on all 26 patients in this study was 22.1, which indicates severe OSA. Post-operative polysomnogram was only performed on 18 patients (69.2%) with an average pre-operative AHI of 21.3 and post-operative AHI of 7.6. Of the 18 patients, all had a decrease in the post-operative AHI, except for one patient who had a slight increase in the post-operative AHI. There was a significant difference (p < 0.001) between the pre- and post-operative AHI of the 18 patients. These results indicate that the surgical treatment that was implemented due to the DISE findings led to AHI improvement and a decrease in the severity of the OSA. Additionally, based on caregiver observation, 12 patients had complete resolution of symptoms and 5 had improvement in OSA symptoms. Post-operative polysomnograms were obtained 3-6 months after the operation. Follow-up of 3-6 months may be short and may not allow for complete observation of surgical outcomes. The studies obtained during that duration, however, did clearly indicate an improvement in post-operative AHI. More longitudinal follow-up would be necessary to determine the persistence of these results.
Thus, based on this series results, DISE could be a solution to the problem of persistent OSA and symptoms after primary adenotonsillectomy. If a patient has risk factors of severe OSA including obesity, African American ethnicity, or medical comorbidities, DISE could be a first line adjunct to treatment. We have demonstrated that the majority of patients that underwent DISE during initial sleep surgery had improvement in symptoms and in post-operative polysomnogram findings. DISE is commonly used second line to determine the cause of non-response to initial adenotonsillectomy [4, 7]. However, with the use of DISE, the knowledge of the site of obstruction would allow the surgeon to implement proper initial surgical treatment targeting the multi-level nature of the obstruction. Physical exam findings related only to adenotonsillar hypertrophy were noted prior to the procedure. Other sites of obstruction would be difficult to determine during routine physical exams due to children often not tolerating flexible laryngoscopy exam while awake. Furthermore, awake endoscopy does not sufficiently detect the site of obstruction in patients with OSA and has been shown to poorly correlate with DISE in adult literature [18]. Therefore, with the use of DISE, a surgeon could better determine additional sites of obstruction compared to a routine physical exam or with an awake flexible laryngoscopy exam. Due to the variety of obstructions that may occur in OSA, in both normal weight and obese children, other surgical options may be preferred for surgical treatment management. Obstruction may be due to a combination of adenotonsillar hypertrophy and nasopharyngeal, retropalatal, retroglossal, and hypopharyngeal obstruction [6]. The most common operations included simultaneous adenotonsillectomy with pharyngolplasty and/or tongue reduction. Personalization of the initial sleep surgery allows for application of early and efficient treatment for OSA. Thus, with timely care, there is an improved chance to reverse the negative consequences of OSA. Additionally, this approach may minimize the number of operations required in the treatment of OSA, thereby decreasing the chance of any adverse effects that may occur due to anesthesia, which inherently carries greater risk in the OSA population. Lastly, there were no reported adverse effects or complications with the usage of DISE. Although increased anesthesia time is an argument against the utility of DISE and subsequent surgery, there were
no observed adverse effects of additional anesthesia time in any of the cases. Furthermore, using DISE at the time of the operation reduced the need for repeated anesthetic administration with future operations. Accordingly, DISE was safe to implement during the initial treatment management of OSA. Although there is less data for post-operative AHI and clinical symptom improvement, the effect of DISE on surgical treatment selection in this cohort of patients resulted in AHI improvement in the majority of patients. A future randomized, controlled study, however, may be of benefit to ensure that DISE, with additional procedures, results in better outcomes than adenotonsillectomy alone. 5. Conclusions DISE has been used as a diagnostic tool for pediatric OSA patients with incomplete response to adenotonsillectomy. We reviewed a group of patients with moderate-severe OSA at risk for persistent OSA following adenotonsillectomy. By incorporating DISE during the initial sleep surgery, the surgeon has an additional tool to assist with intraoperative decision making to efficiently treat multifactorial obstruction. We have shown that it is safe and effective in the pediatric population. Additional patients are needed to confirm these findings and we plan to use these results as the basis for a future long-term randomized controlled trial comparing adenotonsillectomy alone versus DISE plus adenotonsillectomy. Funding source The authors report no funding source. Disclosure of financial information The authors disclose no relevant financial relationships. Informed consent and patient details Retrospective review, exempt study, no informed consent needed.
References [1] D. Gozal, Obstructive sleep apnea in children: implications for the developing central nervous system, Semin. Pediatr. Neurol. 15 (2008) 100-106. https://doi.org/10.1016/j.spen.2008.03.006 [2] C. Guilleminault, J.H. Lee, A. Chan, Pediatric Obstructive Sleep Apnea Syndrome. Arch. Pediatr. Adolesc. Med. 159 (2005) 775–785. https://doi.org/10.1001/archpedi.159.8.775 [3] J. Chan, J. Edman, P.J. Koltai, Obstructive Sleep Apnea, Am. Fam. Physician 69 (2004) 1147-1155. PMID: 15023015 [4] F. Galluzzi, L. Pignataro, R. M. Gaini, W. Garavello, Drug Induced Sleep Endoscopy in the decisionmaking process of children with obstructive sleep apnea. Sleep Med. 16 (2015) 331-335. https://doi.org/10.1016/j.sleep.2014.10.017 [5] A.C. Lin, P.J. Koltai, Sleep endoscopy in the evaluation of pediatric obstructive sleep apnea. Int. J. Pediatr. 2012 (2012) e576719. https://doi.org/10.1155/2012/576719 [6] C.G. Sulman, Pediatric Sleep Surgery. Front. Pediatr. 2 (2014) 51. https://doi.org/10.3389/fped.2014.00051 [7] E.J. Kezirian, Nonresponders to pharyngeal surgery for obstructive sleep apnea: Insights from drug‐ induced sleep endoscopy. The Laryngoscope. 121 (2011) 1320-1326. https://doi.org/10.1002/lary.21749 [8] E. De Corso, A. Fiorita, G. Rizzotto, G.F. Mennuni, D. Meucci, M. Giuliani, et al., The role of druginduced sleep endoscopy in the diagnosis and management of obstructive sleep apnoea syndrome: our personal experience. Acta Otorhinolaryngol. Ital. 33 (2013) 405-13. PMID: 24376297 [9] S.O. Ulualp, P. Szmuk, Drug‐induced sleep endoscopy for upper airway evaluation in children with obstructive sleep apnea. The Laryngoscope. 123 (2013) 292-297. https://doi.org/10.1002/lary.23832
[10] C. Eichler, J.U. Sommer, B.A. Stuck, K. Hormann, J.T. Maurer, Does drug-induced sleep endoscopy change the treatment concept of patients with snoring and obstructive sleep apnea? Sleep Breath. 17 (2013) 63. https://doi.org/10.1007/s11325-012-0647-9 [11] O.M. Vanderveken, Drug-induced sleep endoscopy (DISE) for non-CPAP treatment selection in patients with sleep-disordered breathing. Sleep Breath. 17 (2012) 13-4. https://doi.org/10.1007/s11325012-0671-9 [12] R.A. Franco, R.M. Rosenfeld, M. Rao, Quality of Life for Children with Obstructive Sleep Apnea. Otolaryngol. Head Neck Surg. 123 (2000) 9-16. https://doi.org/10.1067/mhn.2000.105254 [13] I. Narang, J.L. Mathew, Childhood obesity and obstructive sleep apnea. J. Nutr. Metab. 2012 (2012) 134202. https://doi.org/10.1155/2012/134202 [14] K. Nandalike, K. Shifteh, S. Sin, T. Strauss, A. Stakofsky, N. Gonik, et al., Adenotonsillectomy in obese children with obstructive sleep apnea syndrome: magnetic resonance imaging findings and considerations. Sleep 36 (2013) 841-7. https://doi.org/10.5665/sleep.2708 [15] R.B. Mitchell, J. Kelly, Outcome of Adenotonsillectomy for Obstructive Sleep Apnea in Obese and Normal-Weight Children. Otolaryngol Head Neck Surg. 137 (2007) 4348. https://doi.org/10.1016/j.otohns.2007.03.028 [16] C.L. Marcus, R.H. Moore, C.L. Rosen, B. Giordani, S.L. Garetz, H.T. Gerry, et al., A Randomized Trial of Adenotonsillectomy for Childhood Sleep Apnea. N. Engl. J. Med. 368 (2013) 2366-76. https://doi.org/10.1056/NEJMoa1215881. [17] K. El-Kersh, R. Cavallazzi, U.S. Chaddha, E. Senthilvel, Outcomes of adenotonsillectomy in severe pediatric obstructive sleep apnea. Ear Nose Throat J. 96 (2017) E6-E9. https://doi.org/10.1177/014556131709601202.
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Tables
Table 1 Medical comorbidities present in the patient population Medical Comorbidities Number of Patients
Cardiac (23.1%) Arrhythmia Atrial septal defect Patent foramen ovale Dilated right ventricle Cardiorenal syndrome Endocrine (23.1%) Precocious puberty Hyperinsulinemia Acanthosis nigricans Type II diabetes mellitus Hypothyroidism Respiratory (11.5%) Asthma Allergic rhinitis Developmental (11.5 %) Waardenburg syndrome Downs syndrome Speech delay Gastrointestinal (7.7%) Gastroesophageal reflux disease Dysphagia
2 1 1 1 1 1 1 3 2 1 2 1 1 1 2 1 1
Table 2 Drug induced sleep endoscopy findings present in the patient population DISE findings Tonsillar hypertrophy Palatal collapse Base of tongue enlargement Retroflexion of epiglottis Base of tongue collapse Laryngomalacia Lateral pharyngeal collapse Lingual tonsillar hypertrophy Base of tongue obstruction Nasopharynx obstruction
Number of patients 19 17 7 4 3 1 2 2 1 1
Table 3 Individual patient (n=26) pre-operative and post-operative AHI polysomnography results and intervention performed after DISE Polysomnography Pre-operative AHI 20.2 16.4 25.6 11 13.8 23.4 20.2 12.4 23.8 23.5 15.9 26.2 10.7 30.3 13.8 71.8 8.2 14.5 13.8 23.8 43.7 7.2 21.8 45.4 9.7 28.5 T+A= tonsilloadenoidectomy BOT= base of tongue
Intervention T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty, BOT reduction BOT reduction T+A, BOT Reduction T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty
Post-operative AHI 4.3
23.5 15.7 10 1.1 0.8 5.3 1.5 8.6 0.7 25.1 10.7 10.5 0.7 3.8 1.8 5
8.3
Age 6 8 4 9 6 10 8 3 9 4 17 10 4 14 12 4 14 13 12 11 5 6 2 15 3 14
S0165-5876(19)30563-4
DOI:
https://doi.org/10.1016/j.ijporl.2019.109810
Reference:
PEDOT 109810
To appear in:
International Journal of Pediatric Otorhinolaryngology
Received Date: 3 September 2019 Revised Date:
29 November 2019
Accepted Date: 30 November 2019
Please cite this article as: J. Dmowska, S.R. Larson, M.B. Gillespie, A. Sheyn, Effect of Drug Induced Sleep Endoscopy on Intraoperative Decision Making in Pediatric Sleep Surgery International Journal of Pediatric Otorhinolaryngology, https://doi.org/10.1016/j.ijporl.2019.109810. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Elsevier B.V. All rights reserved.
Effect of Drug Induced Sleep Endoscopy on Intraoperative Decision Making in Pediatric Sleep Surgery
Author names and affiliations Julia Dmowska1 Stephen Reed Larson1 M. Boyd Gillespie1 Anthony Sheyn1 1. Department of Otolaryngology, The University of Tennessee Health Science Center, 910 Madison Ave. Memphis, TN 38163; USA Corresponding author Julia Dmowska1 1. Department of Otolaryngology, The University of Tennessee Health Science Center, 910 Madison Ave. Memphis, TN 38163; USA. [email protected].
Conflicts of interest The authors report no conflicts of interest.
Abstract Objectives: To demonstrate the effect of drug induced sleep endoscopy (DISE) on intra-operative decision making during pediatric sleep surgery for obstructive sleep apnea (OSA). Methods: A retrospective chart review was performed on pediatric (3-17 years) patients with moderate-to-severe OSA (7.2-71.8) who underwent drug induced sleep endoscopy at the time of initial sleep surgery. The characteristics evaluated included age, race, gender, site of obstruction, type of surgical intervention, preand post-operative apnea and hypopnea index. Of the 26 patients that were identified, 18 had both a preand post-operative polysomnograms results. Results: All patients underwent DISE immediately prior to surgical treatment. The mean pre-operative AHI for the 18 patients with post-operative polysomnogram results was 21.3 (7.2-71.8). The mean post-operative AHI for the 18 patients was 7.6 (0.7-25.1). There was a significant difference between pre- and post-operative AHI (p < 0.001 ). Of the 26 patients, the most common area of collapse was the soft palate, occurring in 17/26 (65.4%) patients. Base of tongue involvement was found to be present in 11/26 (42.3%) patients, and the epiglottis was involved in 4/26 (15.4%). Evidence of multilevel collapse was observed in 6/26 (23.1%) patients. Patients observed to have palatal collapse underwent a pharyngoplasty (20/26; 76.9%) at the time of adenotonsillectomy. Three (11.5%) patients underwent a tongue reduction.
Conclusion: This study provides additional evidence that DISE can affect intra-operative decision making, with the potential for improved post-operative outcomes. A randomized controlled study is needed to determine if these outcomes are better than what can be achieved without DISE. Keywords Drug induced sleep endoscopy Obstructive sleep apnea Pediatric sleep apnea Adenotonsillectomy
Subdivisions 1. Introduction 2. Materials and methods 3. Results 4. Discussion 5. Conclusions
1. Introduction Obstructive sleep apnea (OSA) is one of the most commonly diagnosed pediatric sleep disorders [1]. Prevalence of this disorder is between 1-3% but may be as high as 5-6% [2]. OSA is associated with repeated episodes of partial or complete airway obstruction during sleep that leads to blood gas abnormalities and sleep fragmentation [1]. This disorder is most commonly associated with adenotonsillar hypertrophy in children [1]. During childhood, the adenoids and tonsils enlarge at a faster rate than the bony structures of the nasopharynx resulting in a narrowed upper airway with increased airflow resistance and recurring upper airway collapse [1]. Adenotonsillar hypertrophy, however, is not always associated with OSA [1]. Additionally, OSA may also be due to other anatomical structures including nasoseptal obstruction, enlarged soft palate or uvula, macroglossia, hypotonic pharynx, micrognathia, maxillary hypoplasia, and lingual tonsil hypertrophy [3]. The current first line treatment, when there is physical evidence of adenotonsillar hypertrophy, is adenotonsillectomy [3]. However, up to 15 to 20% of children have persistent OSA after primary adenotonsillectomy [2, 4, 5]. Persistence of OSA may be due to obesity and/or multifactorial obstruction of the upper airway [5, 6, 7]. Additional surgical options for persistent OSA after adenotonsillectomy are dependent on the site of obstruction and include pharyngoplasty, uvulopalatopharyngoplasty, base of tongue resection, lingual tonsillectomy, septoplasty and supraglottoplasty [6]. Therefore, the ability to identify the site(s) of obstruction prior to an initial surgery would seem to be advantageous to both the patient and surgeon. Drug induced sleep endoscopy (DISE) was introduced in 1991 to provide direct visualization of upper airway collapse under anesthesia [8]. It has been shown to be a safe and useful tool to determine the site, pattern, and degree of upper airway obstruction [5, 9]. Findings include the severity of obstruction in the palatal and hypopharyngeal regions, the collapse of structures including the tongue, epiglottis, velum, and lateral walls, as well as the effect of mouth opening on the airway lumen [7]. In adults, studies have demonstrated that DISE influences choice of therapy and may lead to increased surgical success for the
initial treatment of OSA [8, 10, 11]. DISE may be considered a standard procedure to perform on adults with severe OSA. It has a significant impact on treatment recommendations in adults and guides toward therapeutic options other than CPAP [11]. Informed by the findings on DISE, the surgeon can initiate site specific surgical therapy to treat complex OSA [5]. In pediatrics, DISE is currently used for patients who are non-responsive to initial surgery for OSA [4, 7]. It is largely reserved as a second line diagnostic tool to determine why adenotonsillectomy was not completely effective. One major criticism of DISE in the pediatric population is that it requires additional anesthesia time outside of standard operative management [5]. The current study investigates the utility of DISE during primary sleep surgery for pediatric OSA. The rationale was that DISE would permit observation of upper airway obstruction intra-operatively thereby guiding the initial surgical management. The hope was to achieve improved patient outcomes as defined by post-operative polysomnography with less need for repeated anesthesia. 2. Materials and methods IRB approval was obtained from the University of Tennessee Health Science Center and a retrospective chart review was performed on patients from Le Bonheur Children’s Hospital. Eligible subjects were under 18 years of age at the time of evaluation; met criteria for moderate-severe OSA on a pre-operative polysomnogram with an apnea-hypopnea index (AHI) ≥ 5 and underwent DISE at the time of primary sleep surgery. It was standard practice to consent the care giver of the patient that underwent DISE to include all procedures as “possible.” During the consent process, all families underwent a discussion about the risks and complications of each procedure that could possibly be performed during the time of surgery depending on the findings of the drug induced sleep endoscopy. These procedures included: drug induced sleep endoscopy, adenotonsillectomy, pharyngoplasty, lingual tonsillectomy and base of tongue reduction. Patient and disease related variables were abstracted from the electronic medical records included age, race, gender, BMI, site of obstruction, type of surgical intervention and preand post-operative AHI. During pre-operative examination only adenotonsillar hypertrophy was noted on
physical exam; thus, physical exam findings were not included in the variables reported. Duration of DISE was not specifically measured; the timing was included in the total surgical time. DISE findings were classified at the level of obstruction. Additional surgeries that were performed after DISE depending on findings included: adenotonsillectomy; expansion pharyngoplasty consisting of a tonsillar pillar suture using a vertical mattress suture to connect the posterior tonsillar pillar to the soft palate; and lingual tonsillectomy with midline base of tongue reduction utilizing coblation. This was accomplished by using a Benjamin laryngoscope to expose the base of tongue and using coblation on a setting of 9/3. A pairedsamples t-test was conducted to compare pre-operative and post-operative AHI in patients that had postoperative polysomnogram follow-up. 3. Results A total of 26 patients were identified with documented DISE findings that met the inclusion criteria. The characteristics of the patients were evaluated. The age of the patients ranged from 3 to 17 years old with a mean age of 8.76 (s.d.±4.35). The gender distribution included 8 (30.8%) females and 18 (69.2%) males. Racial background was 22 (84.6%) African Americans, 3 (11.5%) Hispanics and 1 (3.8%) white. Mean BMI was 25.4 (13.9-46.2) with equivalent BMI for age percentile of 75.6% (females 81.9%, males 77.3%). A total of 16/26 (55.1%) patients had medical comorbidities. Six patients had heart conditions (arrhythmia, PFO), 6 had endocrine abnormalities (acanthosis nigricans, hyperinsulinemia, T2DM), 3 had upper airway irregularities (asthma), and 3 had a learning delay (developmental, speech) (Table 1). Sleep endoscopy findings were reviewed (Table 2). The most common area of collapse was the soft palate, occurring in 17 out of 26 patients (65.4%). The base of the tongue was involved in 11 patients (42.3%), with findings of hypertrophy, collapse and obstruction. Retroflexion of the epiglottis was found in 4 patients (15.4%). Tonsillar hypertrophy was found in 19 patients (73.1%). Multilevel collapse was present in 6 out of the 26 patients (23.1%).
Operations were performed for 26 patients (Table 2). All but one patient underwent an adenotonsillectomy (96.2%). Twenty patients (76.9%) underwent a pharyngoplasty at the time of the adenotonsillectomy. Two patients underwent a simultaneous tongue reduction with adenotonsillectomy due to base of tongue hypertrophy and obstruction. One additional patient underwent a tongue reduction operation without adenotonsillectomy. Twenty-six patients had a pre-operative polysomnogram. The average pre-operative AHI was 22.1 (s.d. ±14.0). Post-operative polysomnograms were obtained 3-6 months post-op. A total of 18 patients had post-operative polysomnogram. The average pre-operative AHI, for the patients that had a postoperative polysomnogram results, was 21.3 (sd ±14.3). The average post-operative polysomnogram AHI for the 18 patients was 7.6 (s.d. ±7.4) (Table 3). A paired-samples t-test was conducted to compare preoperative and post-operative AHI of these 18 patients. There was a significant difference in AHI scores (t(17)= 5.05, p < 0.001 ) indicating improvement of AHI after DISE-directed surgery. Eight patients did not have a post-operative polysomnogram. Twenty patients had clinical follow up. Two of the patients with clinical follow up declined a postoperative polysomnogram. At clinical follow up, 12/20 (60.0%) patients had complete resolution of their symptoms according to caregiver confirmation and 5/20 (25.0%) patients had improvement with mild residual symptoms. Two patients (10.0%) had persistent symptoms and one patient (5.9%) had worsening symptomatology which included increased snoring and apneic pauses. No patients required follow-up surgeries. A total of 6/26 (23.1%) patients were lost to follow-up. No adverse events or complications due to the DISE or multi-level airway surgery were reported. No complications occurred due to additional procedures to adenotonsillectomy. Similarly, additional anesthesia time did not adversely affect the patients. 4. Discussion
The patients in this series had many of the recognized risk factors for OSA. Known risk factors include obesity, craniofacial abnormalities, neuromuscular dysfunction, prematurity, Down syndrome, achondroplasia, cerebral palsy and myelomeningocele [1]. Additionally, African American children are more likely than white children to have both OSA and OSA of a more severe degree [1]. Of the charts that were reviewed, 85% of the patients were African American. The most common comorbidities included were cardiac and endocrine in nature. OSA can lead to pulmonary hypertension which can worsen heart disease. Therefore, if heart comorbidities are present, further investigation with DISE into the anatomical cause of the OSA could allow early initiation of treatment to prevent worsening of comorbid conditions. OSA can impair quality of life for children. It may lead to symptoms such as snoring, cessation of breathing, cyanosis and enuresis with consequences of daytime somnolence, behavioral changes, poor academic outcomes, hyperactivity, decline in memory performance and pulmonary hypertension [1, 2, 3, 12]. Early treatment implementation increases the possibility of reversing the complications of OSA [1]. The patients reviewed had OSA symptoms of snoring, daytime somnolence, and to a lesser degree, speech delay. All of these characteristics are consistent with the description of OSA. A total of 17 patients had a BMI percentile greater than 95%. Hence, a majority (65%) of the patients reviewed were obese. Obesity is the most significant risk factor for OSA [1, 13]. Childhood obesity can increase risk for OSA by more than fourfold and OSA can be present in up to 50% of obese children [13, 14]. Obesity is also associated with higher apnea and hypopnea indexes [13]. The American Academy of Pediatrics recommends adenotonsillectomy as the first line treatment for OSA in obese children [14]. Nevertheless, residual OSA will occur in 54-76% of these children [14]. Persistent OSA is more common in obese children compared to normal weight children [15]. Obesity is a profound risk factor for OSA and is associated with residual symptoms after the recommended first line treatment. Based on the characteristics of a patient, especially BMI, it is possible to determine the type of patient that would warrant further surgery, in addition to adenotonsillectomy, to resolve OSA symptoms. These patients
often have multifactorial obstruction and it would be advantageous to use DISE to identify additional sites of obstruction. In this study, patients undergoing DISE were found to have a variety of obstructions of the upper airway. Soft palate collapse, tongue collapse and involvement of the epiglottis were the most common findings outside of adenotonsillar hypertrophy. The current consensus is to perform adenotonsillectomy as the initial operative treatment for OSA [3]. The childhood adenotonsillectomy trial (CHAT) evaluated the efficacy of adenotonsillectomy versus watchful waiting [16]. The trial concluded that there was a greater proportion of normalization of polysomnographic findings in the patients that underwent surgery [16]. Additionally, patients with more severe cases of OSA had larger absolute improvements in polysomnographic findings with surgical intervention [16]. However, there is a recognition that patients with severe OSA are less likely to show normalization compared to less severe cases and that residual disease is very common [5, 6, 7, 16, 17]. The mean AHI on all 26 patients in this study was 22.1, which indicates severe OSA. Post-operative polysomnogram was only performed on 18 patients (69.2%) with an average pre-operative AHI of 21.3 and post-operative AHI of 7.6. Of the 18 patients, all had a decrease in the post-operative AHI, except for one patient who had a slight increase in the post-operative AHI. There was a significant difference (p < 0.001) between the pre- and post-operative AHI of the 18 patients. These results indicate that the surgical treatment that was implemented due to the DISE findings led to AHI improvement and a decrease in the severity of the OSA. Additionally, based on caregiver observation, 12 patients had complete resolution of symptoms and 5 had improvement in OSA symptoms. Post-operative polysomnograms were obtained 3-6 months after the operation. Follow-up of 3-6 months may be short and may not allow for complete observation of surgical outcomes. The studies obtained during that duration, however, did clearly indicate an improvement in post-operative AHI. More longitudinal follow-up would be necessary to determine the persistence of these results.
Thus, based on this series results, DISE could be a solution to the problem of persistent OSA and symptoms after primary adenotonsillectomy. If a patient has risk factors of severe OSA including obesity, African American ethnicity, or medical comorbidities, DISE could be a first line adjunct to treatment. We have demonstrated that the majority of patients that underwent DISE during initial sleep surgery had improvement in symptoms and in post-operative polysomnogram findings. DISE is commonly used second line to determine the cause of non-response to initial adenotonsillectomy [4, 7]. However, with the use of DISE, the knowledge of the site of obstruction would allow the surgeon to implement proper initial surgical treatment targeting the multi-level nature of the obstruction. Physical exam findings related only to adenotonsillar hypertrophy were noted prior to the procedure. Other sites of obstruction would be difficult to determine during routine physical exams due to children often not tolerating flexible laryngoscopy exam while awake. Furthermore, awake endoscopy does not sufficiently detect the site of obstruction in patients with OSA and has been shown to poorly correlate with DISE in adult literature [18]. Therefore, with the use of DISE, a surgeon could better determine additional sites of obstruction compared to a routine physical exam or with an awake flexible laryngoscopy exam. Due to the variety of obstructions that may occur in OSA, in both normal weight and obese children, other surgical options may be preferred for surgical treatment management. Obstruction may be due to a combination of adenotonsillar hypertrophy and nasopharyngeal, retropalatal, retroglossal, and hypopharyngeal obstruction [6]. The most common operations included simultaneous adenotonsillectomy with pharyngolplasty and/or tongue reduction. Personalization of the initial sleep surgery allows for application of early and efficient treatment for OSA. Thus, with timely care, there is an improved chance to reverse the negative consequences of OSA. Additionally, this approach may minimize the number of operations required in the treatment of OSA, thereby decreasing the chance of any adverse effects that may occur due to anesthesia, which inherently carries greater risk in the OSA population. Lastly, there were no reported adverse effects or complications with the usage of DISE. Although increased anesthesia time is an argument against the utility of DISE and subsequent surgery, there were
no observed adverse effects of additional anesthesia time in any of the cases. Furthermore, using DISE at the time of the operation reduced the need for repeated anesthetic administration with future operations. Accordingly, DISE was safe to implement during the initial treatment management of OSA. Although there is less data for post-operative AHI and clinical symptom improvement, the effect of DISE on surgical treatment selection in this cohort of patients resulted in AHI improvement in the majority of patients. A future randomized, controlled study, however, may be of benefit to ensure that DISE, with additional procedures, results in better outcomes than adenotonsillectomy alone. 5. Conclusions DISE has been used as a diagnostic tool for pediatric OSA patients with incomplete response to adenotonsillectomy. We reviewed a group of patients with moderate-severe OSA at risk for persistent OSA following adenotonsillectomy. By incorporating DISE during the initial sleep surgery, the surgeon has an additional tool to assist with intraoperative decision making to efficiently treat multifactorial obstruction. We have shown that it is safe and effective in the pediatric population. Additional patients are needed to confirm these findings and we plan to use these results as the basis for a future long-term randomized controlled trial comparing adenotonsillectomy alone versus DISE plus adenotonsillectomy. Funding source The authors report no funding source. Disclosure of financial information The authors disclose no relevant financial relationships. Informed consent and patient details Retrospective review, exempt study, no informed consent needed.
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Tables
Table 1 Medical comorbidities present in the patient population Medical Comorbidities Number of Patients
Cardiac (23.1%) Arrhythmia Atrial septal defect Patent foramen ovale Dilated right ventricle Cardiorenal syndrome Endocrine (23.1%) Precocious puberty Hyperinsulinemia Acanthosis nigricans Type II diabetes mellitus Hypothyroidism Respiratory (11.5%) Asthma Allergic rhinitis Developmental (11.5 %) Waardenburg syndrome Downs syndrome Speech delay Gastrointestinal (7.7%) Gastroesophageal reflux disease Dysphagia
2 1 1 1 1 1 1 3 2 1 2 1 1 1 2 1 1
Table 2 Drug induced sleep endoscopy findings present in the patient population DISE findings Tonsillar hypertrophy Palatal collapse Base of tongue enlargement Retroflexion of epiglottis Base of tongue collapse Laryngomalacia Lateral pharyngeal collapse Lingual tonsillar hypertrophy Base of tongue obstruction Nasopharynx obstruction
Number of patients 19 17 7 4 3 1 2 2 1 1
Table 3 Individual patient (n=26) pre-operative and post-operative AHI polysomnography results and intervention performed after DISE Polysomnography Pre-operative AHI 20.2 16.4 25.6 11 13.8 23.4 20.2 12.4 23.8 23.5 15.9 26.2 10.7 30.3 13.8 71.8 8.2 14.5 13.8 23.8 43.7 7.2 21.8 45.4 9.7 28.5 T+A= tonsilloadenoidectomy BOT= base of tongue
Intervention T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty, BOT reduction BOT reduction T+A, BOT Reduction T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty T+A T+A, pharyngoplasty T+A, pharyngoplasty T+A, pharyngoplasty
Post-operative AHI 4.3
23.5 15.7 10 1.1 0.8 5.3 1.5 8.6 0.7 25.1 10.7 10.5 0.7 3.8 1.8 5
8.3
Age 6 8 4 9 6 10 8 3 9 4 17 10 4 14 12 4 14 13 12 11 5 6 2 15 3 14