The Laryngoscope C 2013 The American Laryngological, V

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Balloon Laryngoplasty for Pediatric Laryngeal Stenosis: Case Series and Systematic Review Jennifer L. Wentzel, MS; Sidrah M. Ahmad, MD; Christopher M. Discolo, MD, MSCR; M. Boyd Gillespie, MD, MSc; Allison M. Dobbie, MD; David R. White, MD Objectives/Hypothesis: The aim of this study was to systematically review available literature on the outcomes of children treated with balloon laryngoplasty (BLP) as a primary or adjuvant treatment for subglottic or laryngeal stenosis, as well as briefly report on a new series of 60 children treated at the Medical University of South Carolina from 2007 to 2013. Study Design: Review of published case series and retrospective chart review. Methods: A literature search was performed in PubMed and MEDLINE to identify trials that reported clinical outcomes of BLP in human patients under the age of 18 with subglottic or laryngeal stenosis. Single case reports and series studying the dilation of tracheal or bronchial stenosis alone were excluded. Hospital billing codes were used to identify appropriate patients for retrospective chart review. A successful outcome for chart review was determined to be decannulation of previous tracheostomy or avoidance of open laryngotracheoplasty or tracheostomy. Results: Seven studies published between 1991 and 2012 met inclusion criteria and reported outcomes with success defined through improvement of symptoms, decrease in Myer-Cotton level of stenosis, decannulation, or avoidance of reconstructive procedures. Including 60 children from our institution, 202 patients between 1 day and 22 years of age (average 35 months) underwent 457 dilations, with an average of 2.26 dilations per patient (2.25 in our population). The overall success rate was 64% (77% in our population). No complications were reported with subglottic or laryngeal dilations. Conclusions: BLP is a highly effective, low-risk alternative or adjunct to traditional reconstructive procedures in children with subglottic or laryngeal stenosis. Key Words: Balloon, dilation, laryngoplasty, pediatric, children, subglottic stenosis. Level of Evidence: 4 Laryngoscope, 124:1707–1712, 2014

INTRODUCTION Subglottic stenosis (SGS) in children was traditionally considered an infrequent congenital disorder. However, acquired SGS has become increasingly prevalent with advances in airway technologies and the management of premature infants with prolonged endotracheal intubation. The incidence of SGS is estimated at 1% to 2% of intubated neonates1 and 11% of intubated children under 5 years of age, with an increased risk of developing SGS of 50% for every 5 days of intubation.2 Although there are many options available for open surgical reconstruction, endoscopic techniques have the advantage of being less invasive, with the potential of less risk of

From the Department of Otolaryngology–Head and Neck Surgery (J.L.W., C.M.D., M.B.G., A.M.D., D.R.W.), Medical University of South Carolina, Charleston, South Carolina; and Department of Otolaryngology (S.M.A.), Temple University, Philadelphia, Pennsylvania, U.S.A. Editor’s Note: This Manuscript was accepted for publication November 12, 2013. This research will be presented as a poster at the Triological Society Southern Section meeting, Miami Beach, Florida, U.S.A., January 10–12, 2014. The Medical University of South Carolina Institutional Review Board granted approval for this study. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Jennifer L. Wentzel, MS, 135 Rutledge Ave., MSC 550, Charleston, SC 29425. E-mail : [email protected]

infection, shorter hospital stays, and less need for pain management and scar revision. One such form of endoscopic correction of SGS is balloon laryngoplasty (BLP). BLP is a form of endoscopic dilation utilizing an angioplasty balloon first described in 1984 as a method to treat tracheal and bronchial stenoses.3 The advantage of BLP over historical protocols of endoscopic dilation using metal dilators or endotracheal tubes is the ability of the balloon to exert radial pressure that projects the stenosis away from the center of the airway, thereby reducing the incidence of shear-related trauma of the epithelium that may occur with bougienage. BLP has been studied in several limited case series between 1991 and 2012, with populations ranging from 5 to 44 subjects. The aim of this study was to systematically review previous studies to determine the efficacy of BLP on various outcome measures, as well as briefly report on a new series of 60 patients who underwent BLP between 2007 and 2013 at the Medical University of South Carolina Children’s Hospital.

MATERIALS AND METHODS Search Method

DOI: 10.1002/lary.24524

A literature search was performed in PubMed (January 1950 to June 2013) and MEDLINE (January 1966 to June 2013) for studies evaluating the effectiveness of BLP on laryngeal stenosis in children. The key words and Medical Subject

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Headings terms used were “balloon,” AND “dilation,” or “laryngoplasty,” AND “subglottic stenosis,” or “stenosis,” or “intubation,” AND “children,” or “neonates,” or “pediatric.” The only limits used in the search were “humans.” The reference lists of all identified articles were examined for additional relevant studies. All articles were considered regardless of language. The literature search portion of this investigation was considered exempt by the Medical University of South Carolina Institutional Review Board.

Inclusion/Exclusion Criteria Any study that assessed the effectiveness of BLP on clinical outcome measures of congenital or acquired subglottic stenosis in human subjects under the age of 18 was considered for inclusion. One additional study was included that examined BLP in generalized laryngotracheal stenosis. Reviews, single case reports, and series studying the dilation of tracheal or bronchial stenosis alone were excluded.

Case Series A retrospective chart review was conducted through the Medical University of South Carolina. Pertinent patient records were identified by the use of the Common Procedural Terminology (CPT) billing code 31528 for laryngoscopy and dilation. Once the list was generated, medical records were individually reviewed to confirm the use of BLP for laryngotracheal stenosis in a patient under the age of 18 at time of surgery, and to extract demographic and follow-up information, disease type, and severity variables. Cotton-Myer grade of stenosis was determined through chart review or by examining previously recorded laryngoscopy videos.4 Success was determined as decannulation of previous tracheostomy or avoidance of open laryngotracheoplasty or tracheostomy. This study was reviewed and approved by the Medical University of South Carolina Institutional Review Board (HR#17625).

Surgical Technique Balloon dilation was performed under general anesthesia. A 0 bronchoscope was introduced to examine the subglottis, trachea, carina, and mainstem bronchi. The airway was then sized at the point of stenosis using endotracheal tubes. At this point the patient was extubated, and an appropriately sized sinoplasty, esophageal, or cardiac balloon was placed, inflated, and held for a maximum of 2 minutes, assuming maintenance of oxygen saturation. The balloon was deflated and removed and the airway reexamined for patency. Follow-up endoscopy was generally performed 1 week after the procedure. Pre- and postoperative medications and additional procedures varied by case and surgeon preference. The use of topical medications, such as mitomycin, was generally not a part of the treatment algorithm.

RESULTS Included Studies The literature search identified 18 unique abstracts and one additional study found through review of the article reference lists. These 19 records were screened and assessed for eligibility in this review. The search strategy with flow diagram is presented per Preferred Reporting Items for Systematic Reviews and MetaAnalyses guidelines (Fig. 1).5 After review of the 10 prescreened full-length articles, three were excluded due to studying BLP only as an adjunct to other surgical procedures. The seven remaining case series were selected for this review, and results of these studies, as well as the case series reported here, are summarized in Table I.6–12 The seven case series, published between 1991 and 2012, included 142 pediatric patients who underwent a total of 313 dilations, an average 2.26 per patient, for varying degrees of subglottic, tracheal, or laryngeal stenosis. Follow-up ranged from 2 to 24 months, and success was defined as an improvement of symptoms, decrease in Cotton-Myer level of stenosis, decannulation of previous tracheostomy, or avoidance of reconstructive procedures. Rate of success ranged from 54% to 100%, with a total of 93 of the 142 patients (65.5%) reaching a successful outcome. There were no complications reported for subglottic or laryngeal dilations. However, one study reported 11 complications and four deaths, one directly attributable to the procedure, all from attempting to dilate lower tracheal strictures.6 Quality assessment measures were evaluated for the case series as described by Chambers et al.13 Using these criteria, all but one study was considered as having a rating of “poor,” because the data collection was done through retrospective chart review.6–11 The remaining study met all of the Chambers criteria for quality assessment and received a rating of “good.”12

Case Series

Descriptive statistics were generated for the case series data, and successful versus failed cases were compared using v2 or unpaired Student t test with Welch correction, as appropriate. Additionally, bivariate logistic regression analyses were conducted to assess Cotton-Myer grade, number of dilations, age at first procedure, gender, and acquired versus congenital stenosis as potential predictors of BLP failure in SGS. Statistical analysis was performed using GraphPad Prism 6.0 software (GraphPad Software, Inc., La Jolla, CA) and IBM SPSS Statistics for Windows, version 21.0 (IBM, Armonk, NY).

A total of 117 patients were identified in the Medical University of South Carolina database with the billing CPT code 31528. The electronic medical records for these patients were screened, and 57 patients over the age of 18 years were excluded. Of the remaining 60 pediatric patients who underwent a total of 144 balloon dilations, 44 had subglottic stenosis, with the remaining 16 presenting with upper tracheal or laryngeal stenosis through cysts, mucoceles, webs, scar bands, or complete tracheal rings. The age of the population ranged from 4 days to 17 years, with an average of 36.4 months. In 29 cases, BLP was the primary course of treatment and was used as an adjunct to open or endoscopic surgical or laser procedures or tracheotomy in the remaining 31 patients. In cases of congenital stenosis, balloon dilation was used only in the postsurgical management after standard first-line treatment with laryngotracheoplasty or slide tracheoplasty. The 44 SGS patients underwent 93 dilations, and 14 had grade 1 stenosis, eight had grade 2 stenosis, 13 had grade 3 stenosis, and in nine the level of stenosis was unable to be determined

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Fig. 1. Literature search strategy. Preferred Reporting Items for Systematic Reviews and Meta-Analyses flowchart detailing literature search and review.5

through lack of record information. Twenty-seven percent of the SGSs were congenital, and 73% were acquired through prolonged intubation. Average follow-up time was 21.7 6 20 months. Success was achieved in 46 of the 60 total patients, whereas 12 required open surgical laryngotracheal reconstruction, tracheotomy, or maintained a preexisting tracheostomy at their most recent follow-up. One patient underwent surgery within 30 days of chart review, so success or failure was unable to be adequately determined, and one patient was lost to follow-up after having moved out of the area. There were no reported complications.

DISCUSSION

Statistical analyses were performed to assess for potential associations between patient and case characteristics and BLP failures. As depicted in Table II, age at first dilation, gender, the location of the stenosis (subglottic or otherwise), and the primary or adjunctive nature of the dilation were all found to be noncontributory with regard to treatment failure (P >.05). Patients who experienced a failure with BLP underwent significantly more dilations on average than their successful counterparts (P 5.0055). Bivariate analyses of the effects of age, gender, average number of dilations, Cotton-Myer stenosis grade, and whether the dilation was a primary or adjunctive treatment on the odds of treatment failure in SGS yielded no significant correlations (Table III).

Including this series, there have been a total of 457 BLPs performed on 202 pediatric patients for laryngotracheal stenosis reported in the literature between 1991 and 2013. The overall success rate of this procedure is 64%, and no complications have been reported when the procedure was limited to the larynx. Several types of balloon devices have been utilized successfully, and the procedure has proven useful as both a primary and adjunctive therapy in acquired stenosis and as an adjunctive treatment in congenital stenosis after reconstruction is performed. Although the efficacy of balloon dilation as a primary treatment in acquired stenosis is well documented, the utility of BLP in congenital stenosis is less well understood. Two articles and the case series presented here examine the effects of balloon dilation on congenital strictures. Hebra et al. allowed that use of balloon dilation as a primary treatment can possibly temporize until an open reconstruction can be performed.6 Hautefort et al. presented the results of 17 congenital stenosis patients, eight of whom received BLP as a primary treatment, although five of these children underwent LTP more than 1 year previously.10 Of the primary dilations, 50% were successful, compared to 78% of the secondary dilations. In the series presented here, BLP was only done as a secondary measure to laryngoplasty or slide tracheoplasty in congenital cases. Given the amount of force required for a balloon dilation to produce a cricoid fracture, which is the current theory as to how a primary dilation on a congenital stenosis may be

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Wentzel et al.: Balloon Laryngoplasty for Pediatric Stenosis Congenital or acquired laryngotracheal stenosis

Acquired SGS

Congenital (12) or acquired (32) laryngotracheal stenosis

CHD 1 SGS grade III

Acquired SGS with granulation tissue after intubation

SGS (nine) or tracheal stenosis (one), previous LTP failure (six)

SGS secondary to intubation

SGS, tracheal, bronchiol, laryngeal stenosis; congenital or acquired

Disease

N 5 60, 144 dilations, retrospective, F/U 0.8–69 months, average 5 21.7 months

N 5 28, 51 dilations, retrospective, F/U unstated, review over 2-year period

N 5 44, 52 dilations, 31 primary, 21 secondary to recent surgery, retrospective, F/U 1 year

N 5 5, 11 dilations (five in one patient), retrospective, F/U 2–3 months

N 5 8, single dilation per patient, prospective, F/U minimum 6 months

N 5 10, 20 dilations, retrospective, F/U 4–23 months, average 5 10.3 months

N 5 10, 13 dilations, retrospective, F/U 3.5 months

N 5 37, 158 dilations, retrospective, F/U minimum 2 months

Treatment/Follow-up

Avoidance of open reconstruction or tracheostomy, decannulation

Avoidance of open reconstruction or tracheostomy (primary), decannulation (adjuvant)

9/15 (60%) primary dilations successful, 7/13 (53.8%) adjuvant (post-LTP) dilations successful, 9/12 failures in patients with concomitant airway disorders, P 5.048, no complications 46/60 (77%) successful; no association of dilation failure with age, gender, CottonMyer stenosis grade, primary versus adjunct; no complications

All patients avoided immediate postoperative tracheotomy or reconstruction, stenosis resolved in 3/5 (60%), 2/5 underwent LTP a suitable time after CHD surgery 20/31 (65%) primary dilations successful, 17/21 (81%) secondary dilations successful, association of dilation failure with reflux, three or more required dilations and weight

Balloon laryngoplasty for pediatric laryngeal stenosis: case series and systematic review.

The aim of this study was to systematically review available literature on the outcomes of children treated with balloon laryngoplasty (BLP) as a prim...
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