Utility of concurrent direct laryngoscopy and bronchoscopy with drug induced sleep endoscopy in pediatric patients with obstructive sleep apnea

Utility of concurrent direct laryngoscopy and bronchoscopy with drug induced sleep endoscopy in pediatric patients with obstructive sleep apnea

Accepted Manuscript Utility of Concurrent Direct Laryngoscopy and Bronchoscopy with Drug Induced Sleep Endoscopy in Pediatric Patients with Obstructiv...

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Accepted Manuscript Utility of Concurrent Direct Laryngoscopy and Bronchoscopy with Drug Induced Sleep Endoscopy in Pediatric Patients with Obstructive Sleep Apnea Morgan Bliss, Swati Yanamadala, Peter Koltai PII:

S0165-5876(18)30174-5

DOI:

10.1016/j.ijporl.2018.04.009

Reference:

PEDOT 8966

To appear in:

International Journal of Pediatric Otorhinolaryngology

Received Date: 2 October 2017 Revised Date:

8 April 2018

Accepted Date: 10 April 2018

Please cite this article as: M. Bliss, S. Yanamadala, P. Koltai, Utility of Concurrent Direct Laryngoscopy and Bronchoscopy with Drug Induced Sleep Endoscopy in Pediatric Patients with Obstructive Sleep Apnea, International Journal of Pediatric Otorhinolaryngology (2018), doi: 10.1016/j.ijporl.2018.04.009. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. 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.

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Utility of Concurrent Direct Laryngoscopy and Bronchoscopy with Drug Induced Sleep Endoscopy in Pediatric Patients with Obstructive Sleep Apnea Morgan Bliss, MD1

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Swati Yanamadala, MD2 Peter Koltai, MD3

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Department of Otolaryngology-Head and Neck Surgery, Rady Children's Hospital San Diego, University of California-San Diego. 3020 Children’s Way, San Diego, CA 92126. [email protected] 2

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Department of Medicine, Stanford University. 300 Pasteur Drive, Stanford, CA 94305. [email protected]

Department of Otolaryngology-Head and Neck Surgery, Stanford University. 801 Welch Rd, Stanford, CA 94305. [email protected] Potential conflicts of interest: None

Corresponding author:

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Financial disclosures: None

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Morgan Bliss Rady Children's Hospital San Diego/UCSD 3020 Children’s Way, MC 5024 San Diego, CA 92126. Office: 858-309-7701 Email: [email protected]

Key Words: sleep apnea, obstructive; bronchoscopy; child; trachea Accepted for poster presentation at the ASPO 2016 Annual Meeting, May20-22, Chicago, IL.

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Utility of Concurrent Direct Laryngoscopy and Bronchoscopy with Drug Induced Sleep Endoscopy in Pediatric Patients with Obstructive Sleep Apnea

Abstract

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Objectives: The goal of this report was to find the frequency of synchronous airway lesions (SAL) identified during microdirect laryngoscopy and bronchoscopy (MDLB) that influenced treatment decisions beyond the information provided by drug induced sleep endoscopy (DISE)

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alone in children with obstructive sleep apnea (OSA) at a tertiary care pediatric hospital.

Methods: This was a retrospective chart review of all pediatric patients who underwent drug

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induced sleep endoscopy in conjunction with direct laryngoscopy and bronchoscopy as part of a comprehensive airway evaluation for obstructive sleep apnea at a tertiary care pediatric hospital. Results: Three hundred thirty-five patients with obstructive sleep apnea were evaluated with both sleep endoscopy and direct laryngoscopy with bronchoscopy. Five percent of patients had

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SAL identified on MDLB contributing to airway obstruction. Three patients (0.9%) who underwent MDLB for OSA required surgical correction of SAL that was identified. Conclusion: In a limited subset of patients, direct laryngoscopy with bronchoscopy provides

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1. Introduction

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additional positive findings to aid with treatment planning for obstructive sleep apnea.

Obstructive sleep apnea (OSA) is a common condition in childhood, generally caused by

enlarged tonsils and adenoids. However, in children with OSA who have small tonsils and adenoids or who have had adenotonsillectomy, drug induced sleep endoscopy (DISE) has been shown to be an effective technique in identifying sites of obstruction and directing treatment [1]. DISE is often performed concurrently with direct laryngoscopy and bronchoscopy (MDLB),

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however, the incremental value of MDLB over DISE alone for these children remains unclear. The goal of this report was to find the frequency of synchronous airway lesions identified during

children with OSA at a tertiary care pediatric hospital.

2. Methods

After institutional review board (IRB) approval, a retrospective cohort study identified

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MDLB that influenced treatment decisions beyond the information provided by DISE alone in

patients who underwent direct laryngoscopy and bronchoscopy at Lucile Packard Children’s

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Hospital from 2006 through 2014. Operative reports, clinic notes, and sleep studies were reviewed. Both children with refractory OSA after adenotonsillectomy and those with small tonsils and adenoids and no prior surgery were included. Children from 2 months to 18 years of age with obstructive sleep apnea were included in the study. Patients were excluded from the

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study if direct laryngoscopy and bronchoscopy was not performed at the time of surgery.

After performing DISE [2], airway evaluation with MDLB was done. The vocal cords are

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topically anesthetized and a suspended Lindholm laryngoscope is used expose the larynx. A 0-

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degree telescope is then used to inspect the airway while the patient is breathing spontaneously.

3. Results 3.1

Three hundred thirty-five children with obstructive sleep apnea who underwent (MDLB)

concurrently with DISE were identified. Average age at the time of surgery was 7 years. One hundred sixty-two children (48%) had a history of intubation prior to MDLB. This includes one hundred forty-one children who had been intubated only during prior adenotonsillectomy and

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twenty-one children who were intubated for other reasons. Fifty-one children evaluated had an underlying genetic disorder (15%). This includes eighteen children with trisomy 21. Forty-six children were obese (13%), as defined by a body mass index at or above the 95th percentile for

defined by birth prior to 37 weeks gestation.

We included children with other major medical co-morbidities because they are more

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children of the same age. Nineteen were identified as having a history of prematurity (5%), as

likely to have refractory OSA after adenotonsillectomy, and because results of prior studies

or craniofacial problems [3].

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suggest that there may be a larger proportion of SALs in children with neuromuscular disorders

Since our goal was to characterize the incremental value of MDLB over DISE alone in

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identifying SALs, lesions such as laryngomalacia, vocal fold paralysis or paresis, vocal nodules, or enlarged lingual tonsils which were apparent on the DISE were not considered in the results of

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this study.

Synchronous airway lesions (SAL) were identified in 17 patients (5%) during MDLB.

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The most common were subglottic stenosis, subglottic cysts and vascular compression. One of these patients had two lesions identified. Three of the 17 children (18%) had surgical correction of the SAL. Surgical intervention consisted of incision of subglottic cyst with balloon dilation in two patients with subglottic cysts, and balloon dilation in one patient with grade two subglottic stenosis. These results are summarized in Table 1. There were no complications associated with MDLB.

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Thirty-three percent of patients who were intubated for any reason other than

adenotonsillectomy had a SAL identified on MDLB. Only three percent of patients who were

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not intubated or were intubated only during adenotonsillectomy had a SAL identified. Using Fisher’s exact test, history of intubation for any reason other than adenotonsillectomy is

associated with SAL (P < 0.0001). All children with clinically significant and surgically

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correctable lesions did have a history of prior intubation. No patients with SAL identified on

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MDLB had a documented history of stridor on physical exam prior to MDLB.

No children with SAL identified on MDLB were obese. When analyzed individually, neither genetic disorder nor history of prematurity was a predictor for SAL. However, when comparing the rate of SAL in all children with history of prematurity or with genetic, neurologic,

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or craniofacial co-morbidities (19%) to the rate of SAL in otherwise healthy children who were full term (0.4%), there is a significant association between co-morbidity and SAL (P < 0.0001). The average patient age when a SAL was identified on MDLB was 5 years. The average patient

The apnea hypopnea index(AHI) was available for two hundred twenty-five children.

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age when no synchronous airway lesion was found on MDLB was 7 years.

The overall average AHI was 13. The AHI was available for fifteen of the seventeen patients with synchronous airway lesions. The average AHI in this group was 15. The difference in AHI between groups was not statistically significant.

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4. Discussion 4.1

This report highlights a low 5% rate of SALs identified on MDLB, with less than 1% rate

of SALs which require additional surgical correction. Based on these findings, we conclude that

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MDLB in most cases is unnecessary. Our results suggest that concurrent MDLB with DISE may have utility in a limited subset of pediatric patients with OSA who have a history of intubation

genetic, neurologic or craniofacial co-morbidity.

With the increasing use of distal chip fiberoptic scopes for flexible laryngoscopy and

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for any reason other than adenotonsillectomy, or have a history of prematurity, or history of

DISE, it has become easier to visualize lesions of the subglottis during flexible laryngoscopy. This technology may also help surgeons to select patients with OSA who may benefit from further evaluation with MDLB after a potentially concerning lesion is identified on distal chip

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laryngoscopy or DISE.

There are a few limitations to this study. Due to the retrospective nature of the study, it is

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difficult to know for sure whether any of the subglottic lesions identified on MDLB may have also been at least partially visible on DISE. As such, the incremental benefit of concurrent

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MDLB with DISE may be even lower than the findings of this study demonstrate. An additional limitation of this study is that post-operative polysomnograms were not available for some of the children who were found to have SAL on MDLB. Future investigations could compare long term outcomes of children with OSA and SAL who were treated with surgery versus observation alone.

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This is the first study analyzing the marginal benefit of performing MDLB in addition to

DISE specifically in patients with OSA. Previous studies have shown that sleep endoscopy is effective at identifying SAL and plays a significant role in determining the final treatment plan

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[2-7]. In contrast, the current retrospective cohort study shows that less than 1% of patients with difficult to treat OSA had a change in treatment plan due to findings from the MDLB. The

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outcome of this review has changed how we practice.

5. Conclusion

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We conclude that in most cases MDLB concurrently with DISE is unnecessary, except in the subset of patients with history of intubation for reason other than adenotonsillectomy, history

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of prematurity, or history of genetic, neurologic or craniofacial co-morbidity.

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Table 1. Synchronous Airway Lesions Identified During MDLB

6 3 3 2 2 1 1

16% 66% 0% 0% 0% 0% 0%

1.7 3 9.6 1 11.5 12 2

18*

18%

5

Subglottic stenosis Subglottic cyst Vascular compression Tracheobronchomalacia Laryngeal cleft Tracheal stenosis Hypopharyngeal hemangioma

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All SAL identified on MDLB

Comorbidity (number of patients with comorbidity) Prematurity (1), ** Prematurity(2), hydrocephalus, BW Developmental delay DiGeorge Chromosomal translocation

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Required Surgical Intervention

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Frequency

Average Age at Diagnosis (years)

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*One patient in the study had two separate synchronous airway lesions identified on MDLB **BW: Beckwith- Wiedemann

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References [1] Lin AC, Koltai PJ. 2012. International journal of pediatrics 2012; 2012: 576719.

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[2] Truong MT, Woo VG, Koltai PJ. 2012. International journal of pediatric otorhinolaryngology 2012; 76: 722-727.

Pediatric pulmonology 2005; 40: 205-210.

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[3] Goldberg S, Shatz A, Picard E, Wexler I, Schwartz S, Swed E, Zilber L, Kerem E. 2005.

[4] Michelson A, Hawley K, Anne S. Do synchronous airway lesions predict treatment failure

Ped Otolaryngol. 2014; 78: 1439-1443.

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after adenotonsillectomy in children less than 3 years of age with obstructive sleep apnea? Int J

[5] Rastatter J, Schroeder J, French A, Holinger L. Synchronous airway lesions in children younger than age 3 years undergoing adenotonsillectomy. Otolaryngol Head Neck. 2011; 145:

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309-313.

[6] Mandell D, Yellon R. Synchronous airway lesions and esophagitis in children undergoing adenoidectomy. Arch Otolaryngol Head and Neck Surg. 2007; 133:375-380.

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[7]Jaroslava Hybášková, Ondřej Jor, Vilém Novák, Karol Zeleník, PetrMatoušek, and Pavel Ko mínek. Drug-Induced Sleep Endoscopy Changes the Treatment Concept in Patients with

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Obstructive Sleep Apnoea. Bio med Res Int. 2016;2016:6583216.

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Subglottic stenosis Subglottic cyst Vascular compression Tracheobronchomalacia Laryngeal cleft Tracheal stenosis Hypopharyngeal hemangioma All SAL identified on MDLB

Average Required Age at Surgical Diagnosis Comorbidity (number of patients with Frequency Intervention (years) comorbidity) Prematurity (1), 6 16% 1.7 ** 3 66% 3 Prematurity(2), hydrocephalus, BW 3 0% 9.6 Developmental delay 2 0% 1 DiGeorge 2 0% 11.5 Chromosomal translocation 1 0% 12 1 0% 2 Goldenhar 18* 18% 5

*One patient in the study had two separate synchronous airway lesions identified on MDLB

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**BW: Beckwith- Wiedemann