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Injection Augmentation and Endoscopic Repair of Type 1 Isaac et al. Journal of Otolaryngology - Head and Neck Surgery (2020) 49:49 https://doi.org/10.1186/s40463-020-00447-0 ORIGINAL RESEARCH ARTICLE Open Access Injection augmentation and endoscopic repair of type 1 laryngeal clefts: development of a management algorithm Andre Isaac1* , Orysya Svystun2, Wendy Johannsen1,3 and Hamdy El-Hakim1,3 Abstract Objectives: To describe indications for injection augmentation (IA), endoscopic repair (ER) and conservative methods for the management of type 1 laryngeal cleft (LC1) and propose a management algorithm. We also aimed to compare success of IA and ER and determine independent predictors of treatment failure. Methods: Retrospective study of patients diagnosed with LC1 at a Pediatric Otolaryngology referral centre between 2004 and 2016. All had pre-operative instrumental swallowing evaluation (VFSS/FEES), and were managed with a combination of conservative measures, IA and/or ER. We collected demographics, symptoms, comorbidities, VFSS/ FEES results, and operative details. The primary outcome was symptom resolution by parental report. The secondary outcome was predictors of treatment failure. Results: 88 patients were included in the analysis, with mean age 26 ± 25 months. Most presented with choking events (68%) or recurrent pneumonias (48%). In total, there were 55 IA performed and 45 ER. Of the patients who received IA, 19 required subsequent ER. 95% had symptom improvement, 67% had complete resolution. IA had a 56% long-term success rate, whereas that for ER was 85%. Tube feeding at initial evaluation was an independent predictor of treatment failure (HR 11.33 [1.51–84.97], p = 0.018). Conclusions: LC1 can be effectively managed with a combination of IA and ER with favorable results. Failure to respond to IA does not preclude ER, and both have their role in management. Patients who are tube fed have a higher probability of treatment failure. We propose a management algorithm that includes reasoning for conservative approaches, and reduces exposure to general anesthesia. Keywords: Dysphagia, Laryngeal cleft, Swallowing disorders, Pediatrics Introduction accepted classification system for LC was described by Swallowing dysfunction (SD) is a common problem in Benjamin and Inglis in 1989 [1], and includes four types: children and its causes can be complex and multifactor- type 1 LC (LC1) being a dehiscence in the inter- ial. Laryngeal cleft (LC) is an important anomaly that arytenoid muscle resulting in a low inter-arytenoid can be associated with SD and has gained increased at- notch; type 2 (LC2) involving the cricoid cartilage; type tention in the recent literature. The most widely 3 (LC3) extending into the cervical trachea; and type 4 (LC4) involving the intra-thoracic trachea and possibly * Correspondence: [email protected] the carina. LC1 in particular poses a diagnostic challenge 1Pediatric Otolaryngology, Division of Otolaryngology Head & Neck Surgery, [2, 3]. It has been linked with clinical problems such as Departments of Surgery & Pediatrics, The Stollery Children’s Hospital, University of Alberta, Edmonton, Alberta, Canada recurrent aspiration pneumonia, choking during feeding, Full list of author information is available at the end of the article stridor, and failure to thrive. Given that in the last © The Author(s). 2020 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Isaac et al. Journal of Otolaryngology - Head and Neck Surgery (2020) 49:49 Page 2 of 9 decade there has been a surge in the literature on LC1 eligible patients were those children who were evaluated with an increase in diagnosis and management, some au- at the multidisciplinary swallowing clinic at the Stollery thors believe this has been a previously under-diagnosed Children’s Hospital between January and December and under-treated problem [4]. Whilst several authors 2016. We only included children who presented with have advocated their own diagnostic and therapeutic symptoms suggestive of SD, underwent pre-operative in- strategies, a recent consensus guideline from the Inter- strumental swallowing evaluation (videofluoroscopic national Pediatric Otolaryngology working group found swallowing study (VFSS) and/ or functional endoscopic a wide variation in practice among experts [5]. evaluation of swallowing (FEES)) and had a diagnostic Management strategies for LC1 have so far included suspension laryngoscopy and bronchoscopy (SLB) where non-surgical methods such as thickened or modified palpation of the inter-arytenoid area suggested the diag- feeds, alternate route to oral feeding, as well as surgical nosis of LC1. Swallowing evaluation was performed and options including open or endoscopic repair (ER) [6, 7]. interpreted by a certified pediatric Speech and Language More recently, injection augmentation (IA) (also known Pathologist (SLP) in conjunction with a pediatric radi- as injection laryngoplasty) has emerged as an alternative ologist, or a Pediatric Otolaryngologist as per established surgical strategy for LC1 with several authors reporting protocols [14]. Patients for which a baseline VFSS/FEES favourable outcomes [8–12]. However, currently there is was unavailable, those with previous airway surgery, and no consensus in the literature as to the optimal manage- patients with less than three months of follow-up were ment of LC1 patients in terms of threshold and timing excluded. of using various medical and surgical strategies. There is We collected the parameters and variables from the particular controversy around IA, with some authors ad- surgical database, electronic medical records and private vocating its use as a diagnostic and/or prognostic tech- practice records. These included demographic informa- nique [10], whereas others use it as a purely therapeutic tion, presenting symptoms (history of choking and procedure, or a combination of the above [9]. The ma- cough upon feeding and swallowing, stridor, chest infec- jority of the literature did not directly compare IA with tions or pneumonia, apparent life threatening episodes, ER, nor did they have strict criteria of when or why ei- cyanotic spells), prematurity at birth (< 36 week gesta- ther procedure should be pursued. In addition, few au- tion), gastroesophageal reflux disease- GERD (based on thors provided details on how long conservative clinical symptoms, response to proton pump inhibitors), measures should be attempted before proceeding with asthma and/ or atopy (based on clinical history and re- surgery, the role of a single step diagnostic and thera- sponse to bronchodilators, family history), failure to peutic endoscopic examination, and decision-making thrive (<5th WHO percentile for weight or crossing around repeat injections or repairs for patients whose down more than two percentiles consecutively), obesity symptoms recur. There is also variation in practice when (>95th WHO percentile for age and sex), admission to it comes to special populations such as syndromic or intensive care unit with or without intubation, history of neurologically impaired patients, as most studies on the named neurologic impairment, syndromes or dysmorph- topic included a heterogeneous mix of medically com- ism, developmental delay (failure to progress to several plex patients with comorbidities including neurological mile stones) and prior or concomitant surgery. Pre- impairment [9, 10, 12, 13]. Finally, little is known about operative use of an alternate route of feeding (for ex- the parameters of patients who respond to or fail treat- ample nasogastric or gastrostomy tube), as opposed to ment and whether this can be predicted based on pre- oral unmodified or modified intake was recorded. VFSS/ operative variables. FEES data collected included presence of residues, pene- The objective of this study was to describe the indica- tration and/or aspiration (silent or overt). The type of tions of IA, ER and conservative methods for the man- procedure(s) performed was recorded, as well as the re- agement of LC1 through a retrospective review of sponse to treatment and its effect on the route of feeding medically and surgically treated patients, and the cre- or feeds modification. ation of a management algorithm based on our experi- The general outline of management followed in our ence. We also aimed to directly compare the long-term multi-disciplinary clinic was described by Syystun et al. success of IA and ER in achieving symptom resolution, [15]. Largely, where possible a trial of oral thickened li- as well as determine independent predictors of failure
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