Predictive Value of Laryngeal Penetration to Aspiration in a Cohort of Pediatric Patients.

Videofluoroscopic swallow studies (VFSS) provide dynamic assessment of the phases of swallowing under fluoroscopic visualization and allow for identification of abnormalities in the process, such as laryngeal penetration and aspiration. While penetration and aspiration both reflect degrees of swallowing dysfunction, the predictive potential of penetration for subsequent aspiration is not fully elucidated in the pediatric population. As a result, management strategies for penetration vary widely. Some providers may interpret any depth or frequency of penetration as a proxy for aspiration and implement various therapeutic interventions (e.g., modification of liquid viscosity) to eliminate penetration episodes. Some may recommend enteral feeding given the presumed risk of aspiration with penetration, even when aspiration is not identified during the study. In contrast, other providers may advise continued oral feeding without modification even when some degree of laryngeal penetration is identified. We hypothesized that the depth of penetration is associated with the likelihood of aspiration. Identification of predictive factors for aspiration following laryngeal penetration events has significant implications for selection of appropriate interventions. We performed a retrospective cross-sectional analysis of a random sample of 97 patients who underwent VFSS in a single tertiary care center over a 6 month period. Demographic variables including primary diagnosis and comorbidities were analyzed. We examined the association between aspiration and degrees of laryngeal penetration (presence or absence, depth, frequency) across diagnostic categories. Infrequent and shallow penetration events of any type of viscosity were less likely to be associated with aspiration event(s) during the same clinical encounter regardless of diagnosis. In contrast, children with consistent deep penetration of thickened liquids invariably demonstrated aspiration during the same study. Our findings show that shallow, intermittent laryngeal penetration of any viscosity type on VFSS was not consistent with clinical aspiration. These results provide further evidence that penetration-aspiration is not a uniform clinical entity and that nuanced interpretation of videofluoroscopic swallowing findings is necessary to guide appropriate therapeutic interventions.

Pulmonary and Clinical Outcomes After Bilateral Submandibular Gland Excision and Parotid Duct Ligation for Refractory Sialorrhea.

Importance: Refractory sialorrhea in children can result in pulmonary aspiration and irreversible lung damage. Despite many studies devoted to the surgical treatment of sialorrhea, there is a paucity of objective outcome measures after surgery, especially with regard to pulmonary health. Objectives: To assess whether bilateral submandibular gland excision and bilateral parotid duct ligation ("DROOL" procedure) is associated with reduced pulmonary inflammation in bronchoalveolar lavage (BAL) samples after surgery and to assess patient factors associated with improvement after surgery. Design, Setting, and Participants: This retrospective case series included all 112 patients undergoing the DROOL procedure at a single tertiary care pediatric children's hospital from January 1, 2012, to December 31, 2021. Statistical analysis was performed from March 30 to June 10, 2023, and August 20 to September 23, 2023. Exposure: DROOL procedure for refractory sialorrhea. Main Outcomes and Measures: Degree of pulmonary inflammation (neutrophil percentage) according to BAL cytologic findings and overall bronchoscopy findings up to 12 months before and after the DROOL procedure. Secondary outcomes included number of annual hospitalizations, caregiver report of function before and after the procedure, and need for revision procedures and/or additional operations for secretion management. Results: A total of 112 patients (median age, 3.4 years [IQR, 2.0-7.1 years]; 65 boys [58.0%]) underwent DROOL procedures and had both preoperative and postoperative BAL samples during the study period. Patients demonstrated objective improvement in pulmonary inflammation after surgery, with the median polymorphonuclear neutrophil percentage decreasing from 65.0% (IQR, 14.0%-86.0%) before the surgery to 32.5% (IQR, 3.0%-76.5%) after the surgery (median difference in percentage points, -9.0 [95% CI, -20.0 to 0.0]). Prior to the DROOL procedure, 34 patients (30.4%; 95% CI, 21.8%-38.9%) were hospitalized 2 or more times annually for respiratory illness, which decreased to 10.1% (11 of 109; 95% CI, 4.4%-15.7%) after surgery (3 patients did not have hospitalization data available following surgery). Most caretakers (73 [65.2%]) reported improved secretion management after the procedure. Conclusions and Relevance: This study suggests that patients with impaired secretion management who underwent a DROOL procedure demonstrated improvement in pulmonary inflammation and a reduction in hospitalizations after surgery. Caretakers were also likely to report subjective improvement in secretion management and quality of life. Additional research is necessary to guide optimal timing and patient selection for this procedure.

Effects of Vibratory Source on Auditory-Perceptual and Bio-Inspired Computational Measures of Pediatric Voice Quality.

OBJECTIVE: The vibratory source for voicing in children with dysphonia is classified into three categories including a glottal vibratory source (GVS) observed in those with vocal lesions or hyperfunction; supraglottal vibratory sources (SGVS) observed secondary to laryngeal airway injuries, malformations, or reconstruction surgeries; and a combination of both glottal and supraglottal vibratory sources called mixed vibratory source (MVS). This study evaluated the effects of vibratory source on three primary dimensions of voice quality (breathiness, roughness, and strain) in children with GVS, SGVS, and MVS using single-variable matching tasks and computational measures obtained from bio-inspired auditory models. METHODS: A total of 44 dysphonic voice samples from children aged 4-11 years were selected. Seven listeners rated breathiness, roughness, and strain of 1000-ms /ɑ/ samples using single-variable matching tasks. Computational estimates of pitch strength, amplitude modulation filterbank output, and sharpness were obtained through custom-designed MATLAB algorithms. RESULTS: Perceived roughness and strain were significantly higher in children with SGVS and MVS compared to children with GVS. Among the computational measures, only the modulation filterbank output resulted in significant differences among vibratory sources; a posthoc test revealed that children with SGVS had greater amplitude modulation than children with GVS, as expected from their rougher voice quality. CONCLUSIONS: The results indicate that the output of an auditory amplitude modulation filterbank model may capture characteristics of SGVS that are strongly related to the rough voice quality.

A multicenter study analyzing the impact of pre-existing comorbidities on laryngotracheal reconstruction (LTR) outcomes.

INTRODUCTION: Comorbidities such as chronic lung disease and gastroesophageal reflux (GERD), prematurity, and numerous other conditions may impact the success of LTR. Single-center studies are limited in terms of patient numbers and may be underpowered. OBJECTIVES: To analyze the impact of specific comorbidities on the operation-specific and overall surgical success of LTR in a large multicenter cohort and validate a predictive model for surgical success. METHODS: A large retrospective multicenter 10-year review was undertaken to validate the data of a previous single-center study (Wertz et al. Laryngoscope 2020) which identified specific predictive comorbidities which impacted LTR outcomes. A Monte Carlo simulation based on the previous data set suggested that 300-400 cases would be needed to optimize the statistical power of a Bayesian model developed from the single-center data to predict surgical success. An IRB-approved data-sharing agreement was executed for 4 large U.S. CENTERS: A virtual REDCap® data entry form inquired about patient characteristics that best predicted surgical success in the single-center model. These included demographics, surgical approaches, cardiac, airway, genetic, endocrine, musculoskeletal, gastrointestinal, and pulmonary comorbidities; details of the surgical procedures, and results of esophagogastroduodenoscopy (EGD), esophageal pH/impedance and flexible bronchoscopy with bronchioalveolar lavage (BAL) were included. Surgical success defined as successful decannulation or resolution of airway symptoms was recorded as single surgery success and overall success following open surgical revision surgery. Multivariate Bayesian analysis, logistical regression, and Kaplan-Meier analysis were performed. RESULTS: 542 patients were identified, including 165 from the single-center study and an additional 377 patients from the multicenter group. The median age was 36 months at the time of the most recent surgery. 70.9% of the LTRs were double-staged procedures. The overall success rate was 86.4% and operation-specific success rate was 69.2%. The specific comorbidities and aerodigestive test results that impacted success based on univariate analysis included staging, bronchiectasis, pulmonary hypertension, GERD, ASD, PDA, grade of stenosis, advanced levels of stenosis, Trisomy 21, MRSA, prior open surgery at another hospital, and gross appearance on EGD. Bayesian model averaging with backward selection was used to validate and refine a predictive model for surgical success with favorable receiver operating curve characteristics - AUC values of 0.827 for single surgery success and 0.797 for overall success. DISCUSSION: With over 500 patients reviewed, this was the largest multicenter study of LTR to date, which elucidated the impact of comorbidities on success with LTR and was able to improve upon the predictive modeling based on single-center data. Patient factors are most critical in the outcome of LTR. Stage and levels of stenosis, as well as pulmonary and GI conditions most strongly impact the likelihood of success. Future prospective case-control studies will be performed to further optimize the current model for outcome prediction and patient management.

Characteristics and Outcomes of Surgical Treatment for Bronchial Anomalies.

OBJECTIVES: Bronchial anomalies are rare but challenging conditions to treat in children, encompassing a variety of structural abnormalities that could compromise airway patency. This includes complete rings, absent cartilage, traumatic avulsions, bronchoesophageal fistulas, and cartilaginous sleeves. The objective of this study is to describe the characteristics and outcomes of a series of pediatric cases of bronchial anomalies that were treated by slide tracheobronchoplasty. METHODS: This is a single-institution retrospective case series of pediatric patients with bronchial anomalies who underwent surgical treatment between February 2004 and April 2020. Data extracted from electronic medical records included patient demographics, comorbidities, and surgical outcomes. RESULTS: There were a total of 29 patients included in the study, of which 14 had complete bronchial rings, 8 had absent bronchial rings, 4 had traumatic bronchial avulsions, 2 had bronchoesophageal fistulas, and one had a cartilaginous sleeve. Median follow-up time was 13 months (with a range of 0.5-213 months). The overall mortality rate was 17.2% (5 patients), all of whom had complete bronchial rings. Patients with complete bronchial rings also had a higher rate of not only cardiac (85.7%) and pulmonary comorbidities (85.7%) but also secondary airway lesions (78.6%). CONCLUSION: This is the largest series to date describing surgical treatment for bronchial anomalies. Complete bronchial rings were the most common anomaly treated, followed by absent rings and trauma. Surgical treatment can be successful but mortality rates are higher in patients with complete bronchial rings, possibly due to higher rates of pulmonary and cardiac comorbidities. LEVEL OF EVIDENCE: 4 Laryngoscope, 133:3334-3340, 2023.

Revision Endoscopic Posterior Costal Cartilage Grafting: Is It Feasible?

The objective of this work is to examine the feasibility of revision endoscopic posterior costal cartilage graft (EPCCG) placement for posterior glottic stenosis (PGS) and bilateral vocal fold immobility (BVFI). Revision and primary cases were compared with respect to decannulation rates, and it was hypothesized that there would be no difference in outcomes. Twenty-one patients met inclusion criteria (14 primary, 7 revision). Thirteen (62%) had a primary indication of PGS, and 8 (42%) were for BVFI. There were no differences between revision and primary groups with respect to age, gender, or comorbidities (p > .05). There was no difference between groups with respect to decannulation rate (85% primary vs 100% revision, p = .32). Thus, revision EPCCG appears to have comparable results to primary EPCCG with respect to decannulation rate and time to decannulation. EPCCG may be a feasible alternative to open airway reconstruction for PGS and BVFI in selected patients.

Comparative Treatment Outcomes for Idiopathic Subglottic Stenosis: 5-Year Update.

The North American Airway Collaborative (NoAAC) previously published a 3-year multi-institutional prospective cohort study showing variation in treatment effectiveness between 3 primary surgical techniques for idiopathic subglottic stenosis (iSGS). In this report, we update these findings to include 5 years of data evaluating treatment effectiveness. Patients in the NoAAC cohort were re-enrolled for 2 additional years and followed using the prespecified published protocol. Consistent with prior data, prospective observation of 487 iSGS patients for 5 years showed treatment effectiveness differed by modality. Cricotracheal resection maintained the lowest rate of recurrent operation (5%), followed by endoscopic resection with adjuvant medical therapy (30%) and endoscopic dilation (50%). These data support the initial observations and continue to provide value to providers and patients navigating longitudinal decision-making. Level of evidence: 2-prospective cohort study.

Spatial Segmentation for Laryngeal High-Speed Videoendoscopy in Connected Speech.

OBJECTIVE: This study proposes a new computational framework for automated spatial segmentation of the vocal fold edges in high-speed videoendoscopy (HSV) data during connected speech. This spatio-temporal analytic representation of the vocal folds enables the HSV-based measurement of the glottal area waveform and other vibratory characteristics in the context of running speech. METHODS: HSV data were obtained from a vocally normal adult during production of the "Rainbow Passage." An algorithm based on an active contour modeling approach was developed for the analysis of HSV data. The algorithm was applied on a series of HSV kymograms at different intersections of the vocal folds to detect the edges of the vibrating vocal folds across the frames. This edge detection method follows a set of deformation rules for the active contours to capture the edges of the vocal folds through an energy optimization procedure. The detected edges in the kymograms were then registered back to the HSV frames. Subsequently, the glottal area waveform was calculated based on the area of the glottis enclosed by the vocal fold edges in each frame. RESULTS: The developed algorithm successfully captured the edges of the vocal folds in the HSV kymograms. This method led to an automated measurement of the glottal area waveform from the HSV frames during vocalizations in connected speech. CONCLUSION: The proposed algorithm serves as an automated method for spatial segmentation of the vocal folds in HSV data in connected speech. This study is one of the initial steps toward developing HSV-based measures to study vocal fold vibratory characteristics and voice production mechanisms in norm and disorder in the context of connected speech.

Does the History of Tracheoesophageal Fistula Repair Alter Outcomes of Laryngeal Cleft Repair?

OBJECTIVE: Tracheoesophageal fistula and esophageal atresia (TEA) and laryngeal cleft (LC) can coexist in some patients. The surgery-specific success rate of LC repair in children with associated TEA has not been well described. The aim of the study is to determine if the history of TEA alters the LC repair outcomes. STUDY DESIGN: Case series with chart review. SETTING: Single-institution academic medical center. METHOD: A retrospective review was conducted of patients with LC with and without TEA repair between January 2001 and November 2020. Data collected and analyzed included demographics and clinical characteristics, LC type, and LC with TEA timing of repairs. RESULTS: An overall 282 patients met the inclusion criteria of LC repair: LC (n = 242, 85.8%) and LC + TEA (n = 40, 14.2%). Revision repair was required in 43 patients (15.2%) with 8 (2.8%) needing a second revision repair. The first LC revision rate in the LC group was 36/242 (14.9%) as compared with 7/40 (17.5%) in the LC + TEA group (P = .67). The second LC revision rate in the LC and LC + TEA groups was 7 (2.9%) and 1 (2.4%), respectively. The median time to revision was 5.1 months (interquartile range, 3.45-10.6) in the LC group as compared with 29.2 months (interquartile range, 4.8-44.2) in the LC + TEA group (P = .06). CONCLUSION: The incidence of TEA and LC was 14.2% in our study. Based on our findings, history of TEA repair is not associated with a higher revision rate vs LC alone. The history of TEA repair did not alter the outcomes of LC repair.

International Pediatric Otolaryngology Group (IPOG) consensus recommendations: Evaluation and management of congenital tracheal stenosis.

OBJECTIVES: To outline an expert-based consensus of recommendations for the diagnosis and management of pediatric patients with congenital tracheal stenosis. METHODS: Expert opinions were sought from members of the International Pediatric Otolaryngology Group (IPOG) via completion of an 18-item survey utilizing an iterative Delphi method and review of the literature. RESULTS: Forty-three members completed the survey providing recommendations regarding the initial history, clinical evaluation, diagnostic evaluation, temporizing measures, definitive repair, and post-repair care of children with congenital tracheal stenosis. CONCLUSION: These recommendations are intended to be used to support clinical decision-making regarding the evaluation and management of children with congenital tracheal stenosis. Responses highlight the diverse management strategies and the importance of a multidisciplinary approach to care of these patients.

A Deep Learning Approach for Quantifying Vocal Fold Dynamics During Connected Speech Using Laryngeal High-Speed Videoendoscopy.

PURPOSE: Voice disorders are best assessed by examining vocal fold dynamics in connected speech. This can be achieved using flexible laryngeal high-speed videoendoscopy (HSV), which enables us to study vocal fold mechanics with high temporal details. Analysis of vocal fold vibration using HSV requires accurate segmentation of the vocal fold edges. This article presents an automated deep-learning scheme to segment the glottal area in HSV from which the glottal edges are derived during connected speech. METHOD: Using a custom-built HSV system, data were obtained from a vocally healthy participant reciting the "Rainbow Passage." A deep neural network was designed for glottal area segmentation in the HSV data. A recently introduced hybrid approach by the authors was utilized as an automated labeling tool to train the network on a set of HSV frames, where the glottis region was automatically annotated during vocal fold vibrations. The network was then tested against manually segmented frames using different metrics, intersection over union (IoU), and Boundary F1 (BF) score, and its performance was assessed on various phonatory events on the HSV sequence. RESULTS: The designed network was successfully trained using the hybrid approach, without the need for manual labeling, and tested on the manually labeled data. The performance metrics showed a mean IoU of 0.82 and a mean BF score of 0.96. In addition, the evaluation assessment of the network's performance demonstrated an accurate segmentation of the glottal edges/area even during complex nonstationary phonatory events and when vocal folds were not vibrating, thus overcoming the limitations of the previous hybrid approach that could only be applied to the vibrating vocal folds. CONCLUSIONS: The introduced automated scheme guarantees accurate glottis representation in challenging color HSV data with lower image quality and excessive laryngeal maneuvers during all instances of connected speech. This facilitates the future development of HSV-based measures to assess the running vibratory characteristics of the vocal folds in speakers with and without voice disorder. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.19798864.

Surgery and Adjuvant Therapy Improve Derkay Scores in Adult and Pediatric Respiratory Papillomatosis.

OBJECTIVES/HYPOTHESIS: Comparing Derkay anatomical score at time of procedure, disease characteristics, and mean treatment interval among adult and pediatric patients with recurrent respiratory papillomatosis (RRP). STUDY DESIGN: Restrospective study. METHODS: Retrospective review of juvenile-onset (JO) and adult-onset (AO) RRP patients treated longitudinally at pediatric and adult institutions from 1999 to 2019. Patients were included if they had a tissue diagnosis of papilloma and had at least a 12-month follow-up. RESULTS: One hundred and twelve patients met inclusion criteria (68 JO-RRP and 44 AO-RRP). All patients were stratified into either potassium titanyl phosphate (KTP) (n = 42), CO2 (n = 21), or microdebrider (n = 49) treatment groups. The Derkay score improved between first and last procedure in the KTP group (mean difference, 3.5; P < .001), CO2 group (mean difference, 4.4; P < .001), and microdebrider group (mean difference, 4.1; P < .001), but overall improvement did not differ across groups (P = .73). Baseline mean to last mean Derkay score improved for nine patients during bevacizumab treatments (mean difference, 3.0; P = .01) but did not improve for these same patients during an interval prior to receiving bevacizumab treatments. Baseline mean to last mean Derkay score improved for 19 patients during cidofovir treatments (mean difference, 3.84; P < .001) but did not improve for these same patients during the interval prior to receiving cidofovir treatments. The AO-RRP population had more patients with dysplasia (50%) compared to JO-RRP population (10%) (P < .001). CONCLUSION: Various surgical modalities appear to be equally effective treatments for RRP. Adult and pediatric patients have decreased recurrent disease burden when receiving bevacizumab or cidofovir. AO-RRP patients have more concomitant dysplasia. LEVEL OF EVIDENCE: 3 Laryngoscope, 132:2420-2426, 2022.

Consensus on Triple Endoscopy Data Elements Preparatory to Development of an Aerodigestive Registry.

OBJECTIVES/HYPOTHESIS: This study defines essential data elements to be recorded during an aerodigestive "triple endoscopy" to form the foundation of a standardized multicenter registry and to clearly define measurement of each consensus item. STUDY DESIGN: Modified Delphi process. METHODS: Modified Delphi consensus with six survey rounds. Twenty-four expert pediatric otolaryngology, pulmonology, and gastroenterology aerodigestive clinicians from eight large academic pediatric aerodigestive programs formed the Delphi panel. After achieving consensus through the Delphi process, outside validation was performed at 2019 national Aerodigestive Society conference. Consensus, near-consensus, or exclusion was obtained for each proposed data element. Concordance was then measured between expert panel conclusions and validation group conclusions. RESULTS: Overall response rate was 94.4%. 73/167 proposed items reached consensus in six domains (flexible bronchoscopy, bronchoalveolar lavage, microdirect laryngoscopy and bronchoscopy, esophagogastroduodenoscopy with biopsies, and esophageal impedance and pH probe). Measurement of all items was defined; classification/grading systems were selected for 11 items. Validation group endorsed importance of 82/167 data items; compared to expert consensus, overall, inclusion, and exclusion concordance rates were 94.5%, 98.7%, and 90.9%. CONCLUSION: Triple endoscopy is a central component of aerodigestive care. This study identifies and defines data elements to be recorded for all triple endoscopy procedures. The list is of usable length, and clear definitions were created for all items, with explicit classification/grading systems selected for 11 items. Face validity was confirmed with an independent multispecialty sample of aerodigestive providers. This consensus provides the foundation for a triple endoscopy registry but also is immediately applicable to standardize clinical documentation in aerodigestive care. LEVEL OF EVIDENCE: 5 Laryngoscope, 132:2251-2258, 2022.

Perceptual Measures of Boychoir Voices During the Phases of Pubertal Voice Mutation.

BACKGROUND/OBJECTIVES: Vocal changes in the male singing voice associated with puberty are variable and often unpredictable resulting in challenges for the singer and the choral director. Limited knowledge regarding the physiologic changes in the vocal mechanism as they correlate to perceptual variations observed in the male adolescent singer exists in the literature. The purpose of this study was to examine pitch breaks and perceptual characteristics of vocal quality during singing tasks for boys in various stages of the male changing voice. STUDY DESIGN: Prospective Study. METHODS: Twenty-eight boys were initially evaluated at Cooksey Stage 0 (Pubertal Unchanged; n = 15) or Cooksey Stage 1 (Mid-Voice; n = 13). Range of age was 8-13 years old. Participants performed vocal slide intervals (1-3-1, 1-5-1, 1-8-1) with discrete starting frequencies on G3, C4, F4, and A4 and sang the "Star-Spangled Banner" in the key of Ab. Pitch breaks and perceptual qualities were evaluated on the recorded tasks by expert raters. Seven boys were evaluated again when they progressed to Cooksey Stage 4 (Baritone) performing the same singing tasks. RESULTS: For the participants evaluated at Cooksey Stage 0/1, pitch breaks were observed more in the higher frequencies and increased interval spacing regardless of starting frequency. Participants at Cooksey Stage 0 had more pitch breaks than Stage 1. At Cooksey Stage 4, an increase in the number of pitch breaks was observed in comparison to their tasks performed at Stage 0/1 and the perceptual quality of breathiness was significantly greater. CONCLUSIONS: Pitch breaks are a characteristic perceptual change that indicates a young man may be transitioning through puberty. Findings from the present study demonstrate that in addition to perceived pitch breaks, breathiness was noted to significantly increase as the male progressed through puberty. Breathiness was noted to be more significant than vocal timbre and overall vocal quality. This research provides acoustic evidence to enhance the perceptual characteristics of voice change for those who teach and train male voices through puberty.

Quantitative Analysis of Vocal Fold Vibration using High-Speed Videoendoscopy in Children with and without Bilateral Lesions.

OBJECTIVE: To provide data on the measurable vocal fold vibratory differences in children with and without vocal fold lesions using high-speed videoendoscopy. DESIGN: Prospective study, 24 participants (8 healthy; 16 with lesions) between the ages of 5 and 10. METHODS: Rigid high-speed videoendoscopy at the rate of 8,000 frames per second was used to examine participants. Four objective vocal fold phase linearity measures were obtained to establish anterior-posterior contact and separation vibratory patterns. RESULTS: All objective measures showed a difference between nonlesion and bilateral vocal fold lesion groups. Contact-separation patterns in all nonlesion girls and young pre-pubertal boys exhibited an anterior-to-posterior contact and posterior-to-anterior separation; while older boys differed. The objective measures of open quotient, left-right relative phase asymmetry and speed index, showed linear anterior-posterior patterns within the nonlesion group; while the bilateral vocal fold lesion group displayed nonlinear patterns. Patterns in the posterior region of the vocal fold were similar in both groups; while patterns in the anterior region differed. CONCLUSIONS: This study suggests lesions have an effect on the anterior aspect of vocal fold vibratory patterns specifically anterior to the lesions. Age-related differences for males are also evidenced, prompting further investigation of laryngeal development in males and females from childhood to adulthood. This study could serve as a basis for the development of objective clinical measurements of vocal fold vibration in presence of lesions. Further findings could help redefine the theoretical framework of pediatric voice.

Establishing an Endoscopic Chronic Subglottic Stenosis Rabbit Model.

OBJECTIVES/HYPOTHESIS: To develop a reproducible and consistent chronic subglottic stenosis (SGS) in an endoscopic animal model. STUDY DESIGN: Prospective study. METHODS: We conducted a prospective study using New Zealand white rabbits. Chronic SGS was induced endoscopically by Bugbee electrocautery to 50% to 75% of the subglottic area's circumference, followed by 4-hour endotracheal intubation. The rabbit airways were endoscopically assessed and sized with uncuffed endotracheal tubes (ETTs) before the injury, during follow-up, and at the endpoints. There were four endpoints: 2, 4, 6, and 8 weeks post SGS induction. Animals were humanely euthanized for histopathological examination of the subglottic injury site and microscopic measurement of the cricoid lumen. RESULTS: Twenty-two rabbits reached the endpoints, and 18 rabbits developed chronic SGS. ETT size significantly decreased by 0.5 from preinjury to the endpoint in all groups, P < .001. Control median cricoid lumen measurements were 20.48 mm2 , the median cricoid lumen measurement for the 2 weeks endpoint was 14.3 mm2 , 4 weeks 11.69 mm2 , 6 weeks 16.03 mm2 , and 8 weeks endpoint median was 16.33 mm2 . Histopathological examination showed chronic scar tissue and new cartilage formation at the cricoid level, mainly at the posterior subglottic injury site starting from 4 weeks postinjury. Collagen staining revealed substantial amounts of organized collagen and different collagen orientation starting 4 weeks postinjury lasting until 8 weeks postinjury. CONCLUSION: We developed an animal model to study chronic SGS. This model will be utilized to compare different endoscopic treatment interventions in acute SGS versus chronic SGS and further define the molecular basis of SGS. LEVEL OF EVIDENCE: NA Laryngoscope, 132:1909-1915, 2022.

Surgical Treatment of Type III Laryngotracheoesophageal Clefts: Techniques and Outcomes.

OBJECTIVES/HYPOTHESIS: Determine surgical and swallowing outcomes after surgery for type III laryngotracheoesophageal cleft (LTEC). STUDY DESIGN: Case series with chart review. METHODS: Chart review was performed on patients with type III LTEC between 2000 and 2019. Demographics, surgical outcomes, and swallowing outcomes were collected and analyzed. RESULTS: Thirty-three patients met inclusion criteria (28 open and 5 endoscopic repairs). Mean age was 3.4 years for the open group and 0.9 years for the endoscopic group. Seventeen (51.5%) patients had a syndromic diagnosis, most commonly Opitz syndrome and Trisomy 21. Mean follow-up was 33.6 months. Thirteen (39.4%) patients had a previous repair attempt prior to repair at our institution. Twenty-four (70.6%) patients had a tracheostomy prior to or at the time of surgical repair and 13 (38.2%) remain tracheostomy-dependent. Nine patients (27.3%) required a revision cleft repair and four (12.1%) required two revisions. Thirty-one patients had an intact repair at last follow-up (93.9). Two patients died outside the hospital over a year after surgery. Preoperatively 13 of 17 patients with swallowing evaluations aspirated. After repair, 11 of 20 patients were deemed safe for all consistencies and seven were safe for thickened. Endoscopic approaches were performed during the last 2 years of the study and had significantly lower operative time (354.4 minutes vs. 171.5 minutes). CONCLUSIONS: Endoscopic and open approaches are effective for treatment of type III LTEC with 27.3% requiring revision and 93.9% of repairs intact at last follow-up. Overall swallowing outcomes were good in patients who underwent postoperative instrumental swallow evaluation. LEVEL OF EVIDENCE: 4 Laryngoscope, 132:1112-1117, 2022.

Slide Tracheoplasty for Repair of Complex Tracheoesophageal Fistulas.

OBJECTIVES/HYPOTHESIS: Repair of large, recurrent, and complex tracheoesophageal fistulas (TEFs) is challenging and numerous different surgical approaches exist. These various techniques each carry a set of risks and possible complications such as fistula recurrence, tracheal stenosis or pouches, esophageal stenosis, and recurrent laryngeal nerve injury. Slide tracheoplasty is a reconstructive technique successfully used in many different airway pathologies, including TEF repair. This study examines the success, limits, and complications related to slide tracheoplasty for repair of complex TEFs. STUDY DESIGN: Retrospective chart review. METHODS: Patients undergoing TEF repair using a cervical or thoracic approach slide tracheoplasty, at a single institution, between July 2008 and December 2019 were retrospectively reviewed. Demographic data, comorbidities, TEF etiology and surgical history, slide tracheoplasty details and outcomes, and postoperative complication data were examined using descriptive statistics. RESULTS: Twenty-six patients underwent 27 slide tracheoplasties for TEF (20 cervical approaches, 7 thoracic approaches) with a mean age of 5.2 years (IQR 0.7-7.6) at time of surgery. The most common TEF etiologies included congenital (n = 13), tracheostomy tube erosion (n = 5), and button battery ingestion (n = 4). Fistulas ranged in size from <0.5 mm to 4 cm and 59% had previous endoscopic or open repairs. There were two TEF recurrences (7.4%), one of which was successfully revised and the other which was treated with stent placement. Postoperative complications included dehiscence (3.7%), unilateral vocal fold paralysis (3.7%), and mild tracheal stenosis (18.5%). CONCLUSIONS: Slide tracheoplasty is an effective surgical technique for treating complex congenital and acquired TEFs with lower rates of complications when compared to other techniques. LEVEL OF EVIDENCE: 4 Laryngoscope, 132:1542-1547, 2022.

HPV Strain Predicts Severity of Juvenile-Onset Recurrent Respiratory Papillomatosis with Implications for Disease Screening.

Juvenile-onset recurrent respiratory papillomatosis (JoRRP) is the most common benign neoplasm of the larynx in children, presenting with significant variation in clinical course and potential for progression to malignancy. Since JoRRP is driven by human papillomavirus (HPV), we evaluated viral factors in a prospective cohort to identify predictive factors of disease severity. Twenty children with JoRRP undergoing routine debridement of papillomas were recruited and followed for ≥1 year. Demographical features, clinical severity scores, and surgeries over time were tabulated. Biopsies were used to establish a tissue bank and primary cell cultures for HPV6 vs. HPV11 genotyping and evaluation of viral gene expression. We found that patients with HPV11+ disease had an earlier age at disease onset, higher frequency of surgeries, increased number of lifetime surgeries, and were more likely to progress to malignancy. However, the amplitude of viral E6/E7 gene expression did not account for increased disease severity in HPV11+ patients. Determination of HPV strain is not routinely performed in the standard of care for JoRRP patients; we demonstrate the utility and feasibility of HPV genotyping using RNA-ISH for screening of HPV11+ disease as a biomarker for disease severity and progression in JoRRP patients.