Publications by authors named "Evan J Propst"

90 Publications

Synthetic Simulator for Surgical Training in Tracheostomy and Open Airway Surgery.

Laryngoscope 2021 Jan 16. Epub 2021 Jan 16.

Department of Otolaryngology-Head & Neck Surgery, The Hospital for Sick Children, University of Toronto, Toronto, Canada.

Objective(s): To create and validate a synthetic simulator for teaching tracheostomy and laryngotracheal reconstruction (LTR) using anterior costal cartilage and thyroid ala cartilage grafts.

Methods: A late adolescent/adult neck and airway simulator was constructed based on CT scans from a cadaver and a live patient. Images were segmented to create three-dimensional printed molds from which anatomical parts were casted. To evaluate the simulator, expert otolaryngologists - head and neck surgeons performed tracheostomy and LTR using anterior costal cartilage and thyroid ala cartilage grafts on a live anesthetized porcine model (gold standard) followed by the synthetic simulator. They evaluated each model for face validity (realism and anatomical accuracy) and content validity (perceived effectiveness as a training tool) using a five-point Likert scale. For each expert, differences for each item on each simulator were compared using Wilcoxon Signed-Rank tests with Sidak correction.

Results: Nine expert faculty surgeons completed the study. Experts rated face and content validity of the synthetic simulator an overall median of 4 and 5, respectively. There was no difference in scores between the synthetic model and the live porcine model for any of the steps of any of the surgical procedures.

Conclusion: The synthetic simulator created for this study has high face and content validity for tracheostomy and LTR with anterior costal cartilage and thyroid ala cartilage grafts and was not found to be different than the live porcine model for these procedures.

Level Of Evidence: 5 Laryngoscope, 2021.
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http://dx.doi.org/10.1002/lary.29359DOI Listing
January 2021

Suspension Microesophagoscopy for Foreign Body Removal in Children.

Laryngoscope 2020 Nov 6. Epub 2020 Nov 6.

Department of Otolaryngology - Head & Neck Surgery, Hospital for Sick Children, University of Toronto, Toronto, Canada.

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http://dx.doi.org/10.1002/lary.29240DOI Listing
November 2020

Morbidity and mortality from adenotonsillectomy in children with trisomy 21.

Int J Pediatr Otorhinolaryngol 2020 Nov 10;138:110377. Epub 2020 Sep 10.

Department of Otolaryngology - Head and Neck Surgery, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada. Electronic address:

Objective: Adenotonsillectomy (AT) is common in children with trisomy 21 but outcomes are variable. Therefore, practitioners must have accurate information regarding the risks of the procedure specific to trisomy 21 to help patients weigh the risks and benefits of surgery. The objective of this study was to better characterize morbidity and mortality risk factors from AT in children with trisomy 21.

Methods: A single-center retrospective chart review of children with trisomy 21 who underwent AT was conducted from 1992 to 2019. The primary outcome was 30-day post-operative complication rate. Secondary outcomes included intraoperative complications, admission duration, emergency department visits, readmissions, reoperation rate and treatment failures.

Results: Two-hundred and fifty one children met study criteria (median age 4.5 years). Seventy-eight patients (31.5%) had a post-operative complication requiring medical intervention, with respiratory issues (42, 53.8%), poor oral intake (29, 37.2%), and bleeding (14, 17.9%) being most common. Postoperatively, 72 patients (28.7%) had a prolonged hospital stay. Sleep disordered breathing (p = 0.003), ASA score >2 (p < 0.001), severe OSA (p = 0.003), preoperative ICU admission (p < 0.001), and aerodigestive comorbidities (p = 0.004) were associated with increased post-operative respiratory complications. No mortalities were identified.

Conclusion: This large single institution study evaluating morbidity and mortality following AT in children with trisomy 21 identified a morbidity rate of 31.5%. These findings may improve our ability to anticipate and manage postoperative morbidity in this vulnerable population and facilitate informed discussions with patients and caregivers considering AT.
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http://dx.doi.org/10.1016/j.ijporl.2020.110377DOI Listing
November 2020

International Pediatric Otolaryngology Group (IPOG) consensus recommendations: Management of suprastomal collapse in the pediatric population.

Int J Pediatr Otorhinolaryngol 2020 Dec 13;139:110427. Epub 2020 Oct 13.

Children's Health Ireland (Crumlin), Dublin, Ireland.

Introduction: Suprastomal Collapse (SuStCo) is a common complication of prolonged tracheostomy in children. There is a paucity of literature on this subject, especially regarding how to manage significant suprastomal collapse that prevents safe decannulation.

Objective: Provide a definition, classification system, and recommend management options for significant suprastomal collapse in children with tracheostomy.

Methods: Members of the International Pediatric Otolaryngology Group (IPOG) who are experts in pediatric airway conditions were surveyed and results were refined using a modified Delphi method.

Results: Consensus was defined as > 70% agreement on a subject. The experts achieved consensus: CONCLUSION: This consensus statement provides recommendations for medical specialists who manage infants and children with tracheostomies with significant Suprastomal Collapse. It provides a classification system to facilitate diagnosis and treatment options for this condition.
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http://dx.doi.org/10.1016/j.ijporl.2020.110427DOI Listing
December 2020

Current management of type III and IV laryngotracheoesophageal clefts: the case for a revised cleft classification.

Curr Opin Otolaryngol Head Neck Surg 2020 Dec;28(6):435-442

University of Washington, Department of Otolaryngology-Head & Neck Surgery and Seattle Children's Hospital, Seattle, Washington, USA.

Purpose Of Review: This review summarizes the paediatric laryngotracheoesophageal cleft (LTEC) literature, with an emphasis on recent trends, evaluation and management, surgical techniques, postoperative care of Type III and IV LTECs, and to propose a revised cleft classification system that more accurately reflects our current understanding of these anomalies.

Recent Findings: There are a number of techniques described to address Type III and IV LTEC, from endoscopic to open approaches with thoracotomy. The surgical approach should be tailored to the length of the cleft and its proximity to important anatomical structures. On the basis of review of the literature, we propose a modified Benjamin-Inglis classification (MBI) with subcategories to address this issue. Postoperative complications are common, namely, tracheoesophageal fistulae and tracheomalacia, which may necessitate subsequent procedures or prolonged tracheostomy dependence.

Summary: The medical and surgical management of Type III and IV LTEC is challenging with a high rate of morbidity and mortality. The rarity and difficulties in management of these malformations have made large cohort studies difficult, thus generalizable recommendations have been elusive. Experience and patient selection are critical for successful endoscopic repair. Anterior cervical approach, often with complete laryngofissure, appears to be the most common and preferred method for open repairs, though some use a lateral approach. The proposed MBI classification appears to be a useful adjunct to aid in surgical decision-making for deeper LTEC.
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http://dx.doi.org/10.1097/MOO.0000000000000669DOI Listing
December 2020

Systematic review of rigid and flexible esophagoscopy for pediatric esophageal foreign bodies.

Int J Pediatr Otorhinolaryngol 2020 Dec 25;139:110397. Epub 2020 Sep 25.

Department of Otolaryngology - Head & Neck Surgery, University of Toronto, Toronto, ON, Canada; Division of Otolaryngology - Head & Neck Surgery, Trillium Health Partners, Mississauga, ON, Canada. Electronic address:

Background: Esophageal foreign body (EFB) is a common presentation in pediatric hospitals. Rigid and flexible esophagoscopy are both primary modalities used for EFB removal. By systematically reviewing the literature, we sought to assess success and complication rates of both rigid and flexible esophagoscopy in pediatric EFB retrieval.

Methods: A systematic search of the literature was performed in MEDLINE, Embase, Cochrane Database of Systematic Reviews, Web of Science, CINAHL. Two authors independently reviewed 74 abstracts and included studies that documented success rates of flexible and rigid esophagoscopy for EFB retrieval. The certainty and quality of the overall body of evidence was evaluated in accordance with the Grades of Recommendation, Assessment, Development and Evaluation approach.

Results: Five retrospective chart reviews published between 1993 and 2016 identified 955 patients with a mean age of 4.2 (range 3.2-5.3 years). Foreign bodies were localized to the upper esophagus in 69.6%, middle esophagus in 13.0% and lower esophagus in 11.5%. Coins (82.1%) and food boluses (8.5%) comprised the majority of EFB. The success rate of foreign body retrieval was 95.4% with rigid esophagoscopy and 97.4% with flexible esophagoscopy. Complication rates were 2.4% for rigid esophagoscopy and 3.4% for flexible esophagoscopy. Rates of successful foreign body retrieval (odds ratio [OR] = 1.9 [95% CI 0.9-3.8; p = 0.07]) and complications (OR = 0.9 [95% CI 0.3-2.3; p = 0.79]) were not significantly different across modalities. There were no mortalities in any of the studies analyzed.

Conclusion: Flexible and rigid esophagoscopy are equally safe and effective for the retrieval of EFB in children. Otolaryngologists, gastroenterologists, and pediatric general surgeons can be consulted for foreign body retrieval depending on institutional policies and resource availability.
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http://dx.doi.org/10.1016/j.ijporl.2020.110397DOI Listing
December 2020

Competency-Based Assessment Tool for Pediatric Esophagoscopy: International Modified Delphi Consensus.

Laryngoscope 2020 Oct 9. Epub 2020 Oct 9.

Department of Otolaryngology - Head and Neck Surgery, Hospital for Sick Children, University of Toronto, Toronto, Canada.

Objectives/hypothesis: Create a competency-based assessment tool for pediatric esophagoscopy with foreign body removal.

Study Design: Blinded modified Delphi consensus process.

Setting: Tertiary care center.

Methods: A list of 25 potential items was sent via the Research Electronic Data Capture database to 66 expert surgeons who perform pediatric esophagoscopy. In the first round, items were rated as "keep" or "remove" and comments were incorporated. In the second round, experts rated the importance of each item on a seven-point Likert scale. Consensus was determined with a goal of 7 to 25 final items.

Results: The response rate was 38/64 (59.4%) in the first round and returned questionnaires were 100% complete. Experts wanted to "keep" all items and 172 comments were incorporated. Twenty-four task-specific and 7 previously-validated global rating items were distributed in the second round, and the response rate was 53/64 (82.8%) with questionnaires returned 97.5% complete. Of the task-specific items, 9 reached consensus, 7 were near consensus, and 8 did not achieve consensus. For global rating items that were previously validated, 6 reached consensus and 1 was near consensus.

Conclusions: It is possible to reach consensus about the important steps involved in rigid esophagoscopy with foreign body removal using a modified Delphi consensus technique. These items can now be considered when evaluating trainees during this procedure. This tool may allow trainees to focus on important steps of the procedure and help training programs standardize how trainees are evaluated.

Level Of Evidence: 5. Laryngoscope, 2020.
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http://dx.doi.org/10.1002/lary.29126DOI Listing
October 2020

Positional obstructive sleep apnea in an obese pediatric population.

J Clin Sleep Med 2020 08;16(8):1295-1301

Translational Medicine, Research Institute, Hospital for Sick Children, Toronto, Canada.

Study Objectives: Positional obstructive sleep apnea (POSA) is a phenotype of obstructive sleep apnea (OSA) where sleep-related obstructive events occur predominantly in the supine position. Limited knowledge exists regarding the presence of POSA in children with obesity. The study objective was to determine the prevalence of POSA while identifying factors associated with POSA in children with obesity.

Methods: This was a cross-sectional study of children with obesity, aged 8 to 18 years, with a diagnostic polysomnogram (PSG) between 2012 to 2019, who were referred for the evaluation of sleep-related breathing. POSA was defined as an overall obstructive apnea-hypopnea index (OAHI) ≥5 events/h and a supine OAHI to nonsupine OAHI ratio of ≥2. Patient demographics, anthropometrics, and PSG data were recorded.

Results: Of the 112 children with obesity with a diagnostic PSG, 43 (38%) had OSA. Among those with OSA, 25 of 43 (58%) had POSA (mean age: 14.6 ± 2.3 years; mean body mass index: 37.7 ± 7.6 kg/m²; 68% male) and 18 of 43 (42%) had non-POSA (mean age: 13.9 ± 2.8 years; mean body mass index: 37.9 ± 7.2 kg/m²; 78% male). Among those with POSA, 13 of 25 (52%) had mild OSA, 7 of 25 (28%) had moderate OSA, and 5 of 25 (20%) had severe OSA. No significant differences were found in age, sex, and anthropometric measures between POSA and non-POSA groups. Time spent in supine and nonsupine sleep did not differ significantly between groups.

Conclusions: In children with obesity and OSA, POSA occurs frequently. Identifying POSA allows for potential targeted positional therapy for children with obesity.
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http://dx.doi.org/10.5664/jcsm.8496DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7446091PMC
August 2020

Fetoscopic Balloon Dilation and Cricotracheal Resection for Laryngeal Atresia in CHAOS.

Laryngoscope 2021 03 23;131(3):660-662. Epub 2020 Jun 23.

University of Toronto, Toronto, Ontario, Canada.

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http://dx.doi.org/10.1002/lary.28775DOI Listing
March 2021

FCRL4 Is an Fc Receptor for Systemic IgA, but Not Mucosal Secretory IgA.

J Immunol 2020 Jul 8;205(2):533-538. Epub 2020 Jun 8.

Department of Immunology, University of Toronto, Toronto, Ontario M5S 1A8, Canada;

Fc receptor-like (FCRL) 4 is an immunoregulatory receptor expressed on a subpopulation of human memory B cells of mucosa-associated lymphoid tissue. Fc receptor function of FCRL4 was demonstrated by binding of IgA to FCRL4 following heat aggregation of the Ig. In this study, we demonstrate that FCRL4 recognizes J chain-linked systemic IgA in the absence of heat aggregation. We further demonstrate that mucosal secretory IgA is not recognized by FCRL4 and that systemic IgA binding can be competitively inhibited by recombinant secretory component protein. Finally, we provide evidence that primary FCRL4-bearing human memory B cells are constitutively bound to IgA. Our study provides a mechanism for the negative regulatory activity of FCRL4 on AgR-mediated B cell activation.
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http://dx.doi.org/10.4049/jimmunol.2000293DOI Listing
July 2020

Pediatric laryngoscopy and bronchoscopy during the COVID-19 pandemic: A four-center collaborative protocol to improve safety with perioperative management strategies and creation of a surgical tent with disposable drapes.

Int J Pediatr Otorhinolaryngol 2020 Jul 21;134:110059. Epub 2020 Apr 21.

Department of Otolaryngology, Head and Neck Surgery, University of Toronto, The Hospital for Sick Children, Toronto, Canada.

Aerosolization procedures during the COVID-19 pandemic place all operating room personnel at risk for exposure. We offer detailed perioperative management strategies and present a specific protocol designed to improve safety during pediatric laryngoscopy and bronchoscopy. Several methods of using disposable drapes for various procedures are described, with the goal of constructing a tent around the patient to decrease widespread contamination of dispersed droplets and generated aerosol. The concepts presented herein are translatable to future situations where aerosol generating procedures increase risk for any pathogenic exposure. This protocol is a collaborative effort based on knowledge gleaned from clinical and simulation experience from Children's Hospital Colorado, Children's Hospital of Philadelphia, The Hospital for Sick Children in Toronto, and Boston Children's Hospital.
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http://dx.doi.org/10.1016/j.ijporl.2020.110059DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7172675PMC
July 2020

Consensus treatment plans for periodic fever, aphthous stomatitis, pharyngitis and adenitis syndrome (PFAPA): a framework to evaluate treatment responses from the childhood arthritis and rheumatology research alliance (CARRA) PFAPA work group.

Pediatr Rheumatol Online J 2020 Apr 15;18(1):31. Epub 2020 Apr 15.

Joseph M Sanzari Children's Hospital, Hackensack Meridian Health, Hackensack, NJ, USA.

Background: Periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis (PFAPA) syndrome is the most common periodic fever syndrome in children. There is considerable heterogeneity in management strategies and a lack of evidence-based treatment guidelines. Consensus treatment plans (CTPs) are standardized treatment regimens that are derived based upon best available evidence and current treatment practices that are a way to enable comparative effectiveness studies to identify optimal therapy and are less costly to execute than randomized, double blind placebo controlled trials. The purpose of this project was to develop CTPs and response criteria for PFAPA.

Methods: The CARRA PFAPA Working Group is composed of pediatric rheumatologists, infectious disease specialists, allergists/immunologists and otolaryngologists. An extensive literature review was conducted followed by a survey to assess physician practice patterns. This was followed by virtual and in-person meetings between 2014 and 2018. Nominal group technique (NGT) was employed to develop CTPs, as well as inclusion criteria for entry into future treatment studies, and response criteria. Consensus required 80% agreement.

Results: The PFAPA working group developed CTPs resulting in 4 different treatment arms: 1. Antipyretic, 2. Abortive (corticosteroids), 3. Prophylaxis (colchicine or cimetidine) and 4. Surgical (tonsillectomy). Consensus was obtained among CARRA members for those defining patient characteristics who qualify for participation in the CTP PFAPA study.

Conclusion: The goal is for the CTPs developed by our group to lead to future comparative effectiveness studies that will generate evidence-driven therapeutic guidelines for this periodic inflammatory disease.
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http://dx.doi.org/10.1186/s12969-020-00424-xDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7157990PMC
April 2020

Detection of Human CD38 Using Variable Lymphocyte Receptor (VLR) Tetramers.

Cells 2020 04 12;9(4). Epub 2020 Apr 12.

Department of Immunology, University of Toronto, Toronto, ON M5S 1A1, Canada.

CD38 is a multifunctional cell surface receptor expressed on multiple cell lineages of hematopoietic origin with high levels of expression on human plasma cells. Previously, we isolated the monoclonal variable lymphocyte receptor B (VLRB) MM3 antibody from the evolutionarily distant sea lamprey, which recognized the CD38 ectoenzyme exclusively on human plasma cells in a manner that correlated with CD38 enzymatic activity. The plasma cell-specific binding of VLRB MM3 contrasts with the broad pattern of expression of CD38-determined conventional antibodies specific for this antigen. In an effort to facilitate the application of this unique reagent in combination with conventional antibody panels, we explored a strategy to generate VLRB MM3 tetramers. The resulting reagent maintained the threshold-based recognition of CD38. Increased sensitivity achieved with VLRB MM3 tetramers also showed preferential recognition of germinal center centroblasts over centrocytes. VLRB MM3 tetramers thus provided a unique and versatile single-step staining reagent for the detection of human CD38 that is readily incorporated into multi-color flow cytometry panels.
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http://dx.doi.org/10.3390/cells9040950DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7226959PMC
April 2020

Positional obstructive sleep apnea in an obese pediatric population.

J Clin Sleep Med 2020 Apr 14. Epub 2020 Apr 14.

Translational Medicine, Research Institute, Hospital for Sick Children, Toronto, Canada.

Study Objectives: Positional obstructive sleep apnea (POSA) is a phenotype of obstructive sleep apnea (OSA) where sleep-related obstructive events occur predominantly in supine position. Limited knowledge exists regarding the presence of POSA in children with obesity. The study objective was to determine the prevalence of POSA while identifying factors associated with POSA in children with obesity.

Methods: This was a cross-sectional study of children with obesity, aged 8 to 18 years, with a diagnostic polysomnogram (PSG) between 2012 to 2019, who were referred for the evaluation of sleep-related breathing. POSA was defined as an overall obstructive apnea-hypopnea index (OAHI) ≥ 5 events per hour, and a supine OAHI to non-supine OAHI ratio of ≥2. Patient demographics, anthropometrics, and PSG data were recorded.

Results: Of the 112 children with obesity with a diagnostic PSG, 43 (38%) had OSA. Amongst those with OSA, 25/43 (58%) had POSA (mean age 14.6±2.3 years, mean BMI 37.7±7.6 kg/m², 68% male) and 18/43 (42%) had no-POSA (mean age 13.9 ± 2.8 years, mean BMI 37.9 ± 7.2 kg/m², 78% male). Among those with POSA, 13/25 (52%) had mild OSA, 7/25 (28%) had moderate OSA and 5/25 (20%) had severe OSA. No significant differences were found in age, gender and anthropometric measures between POSA and no-POSA groups. Time spent in supine and non-supine sleep did not differ significantly between groups.

Conclusions: In children with obesity and OSA, POSA occurs frequently. Identifying POSA allows for potential targeted positional therapy for children with obesity.
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April 2020

Diagnosis and management of complete tracheal rings with concurrent tracheoesophageal fistula.

Int J Pediatr Otorhinolaryngol 2020 Jun 27;133:109971. Epub 2020 Feb 27.

Department of Otolaryngology - Head and Neck Surgery, The Hospital for Sick Children, University of Toronto, Toronto, Canada.

Objective: Characterize patients with complete tracheal rings and tracheoesophageal fistula (TEF) and summarize management options.

Methods: A systematic review of patients under 18 years of age with complete tracheal rings and TEF was conducted. Authors were contacted for additional patient information and new cases were added. Patients with iatrogenic TEF and tracheal stenosis due to other causes were excluded.

Results: Sixteen patients with a median (IQR) follow-up of 10 months (3-12 months) were identified. All had a distal TEF with complete tracheal rings distal to the TEF. There were 10 (63%) type C esophageal atresia + TEF (EA/TEF), and 1 (6%) type D (5 missing data). Median (IQR) airway diameter was 2 mm (1.5-2.2 mm). Complete tracheal rings were diagnosed prior to TEF repair in 5 (31.3%) patients, after ≥1 failed extubation in 3 (12.5%) patients, and intra-operatively during respiratory distress in 1 patient. Ten patients (62.5%) were intubated with an endotracheal tube and one with a 6 Fr flexible aortic canula (5 missing data). Four patients with an endotracheal tube for TEF repair developed ventilatory problems. Complete tracheal rings were repaired in 9 (56%) patients (8 slide tracheoplasty, 1 pericardial patch) and followed conservatively in 3 (19%). One patient required tracheotomy. Four patients died.

Conclusions: Complete tracheal rings with concurrent TEF is a rare entity that pose challenges for ventilatory management during operative repair. Bronchoscopy prior to TEF repair is critical to allow for proper preoperative planning.
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http://dx.doi.org/10.1016/j.ijporl.2020.109971DOI Listing
June 2020

Development of a Modern Pediatric Airway Reference Tool.

Laryngoscope 2020 10 30;130(10):2428-2431. Epub 2019 Dec 30.

Department of Otolaryngology-Head & Neck Surgery, University of Washington, Seattle, Washington, U.S.A.

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http://dx.doi.org/10.1002/lary.28437DOI Listing
October 2020

Competency-Based Assessment Tool for Pediatric Tracheotomy: International Modified Delphi Consensus.

Laryngoscope 2020 11 10;130(11):2700-2707. Epub 2019 Dec 10.

Department of Pediatric Otolaryngology, Phoenix Children's Hospital, Phoenix, Arizona, U.S.A.

Objectives/hypothesis: Create a competency-based assessment tool for pediatric tracheotomy.

Study Design: Blinded, modified, Delphi consensus process.

Methods: Using the REDCap database, a list of 31 potential items was circulated to 65 expert surgeons who perform pediatric tracheotomy. In the first round, items were rated as "keep" or "remove," and comments were incorporated. In the second round, experts were asked to rate the importance of each item on a seven-point Likert scale. Consensus criteria were determined a priori with a goal of 7 to 25 final items.

Results: The first round achieved a response rate of 39/65 (60.0%), and returned questionnaires were 99.5% complete. All items were rated as "keep," and 137 comments were incorporated. In the second round, 30 task-specific and seven previously validated global rating items were distributed, and the response rate was 44/65 (67.7%), with returned questionnaires being 99.3% complete. Of the Task-Specific Items, 13 reached consensus, 10 were near consensus, and 7 did not achieve consensus. For the 7 previously validated global rating items, 5 reached consensus and two were near consensus.

Conclusions: It is feasible to reach consensus on the important steps involved in pediatric tracheotomy using a modified Delphi consensus process. These items can now be considered to create a competency-based assessment tool for pediatric tracheotomy. Such a tool will hopefully allow trainees to focus on the important aspects of this procedure and help teaching programs standardize how they evaluate trainees during this procedure.

Level Of Evidence: 5 Laryngoscope, 130:2700-2707, 2020.
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http://dx.doi.org/10.1002/lary.28461DOI Listing
November 2020

Live porcine model for surgical training in tracheostomy and open-airway surgery.

Laryngoscope 2020 08 30;130(8):2063-2068. Epub 2019 Sep 30.

Department of Otolaryngology-Head and Neck Surgery, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.

Objectives/hypothesis: To evaluate the validity of a live porcine model for surgical training in tracheostomy and open-airway surgery.

Study Design: Prospective observational study.

Methods: Eleven expert otolaryngologists-head and neck surgeons rated a live porcine model's realism/anatomical accuracy (face validity) and perceived effectiveness as a training tool (content validity) for tracheostomy and laryngotracheoplasty using anterior costal cartilage and thyroid ala cartilage grafts using a 53-item post-trial questionnaire with a five-point Likert scale.

Results: Experts rated the face validity of the live porcine model a median (interquartile range [IQR]) of 4/5 (4-5) and the content validity a median (IQR) of 5/5 (4-5) for each surgical procedure. Overall, 91% strongly agreed or agreed that the simulator would increase trainee competency for tracheostomy and laryngotracheoplasty using costal cartilage graft, and 82% strongly agreed or agreed that it would increase trainee competency for laryngotracheoplasty using thyroid ala cartilage graft.

Conclusions: The live porcine model has high face and content validity as a training tool for tracheostomy and laryngotracheoplasty using costal cartilage and thyroid ala cartilage grafts. This training model can help surgical trainees practice these complex, low-frequency procedures.

Level Of Evidence: NA Laryngoscope, 130: 2063-2068, 2020.
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http://dx.doi.org/10.1002/lary.28309DOI Listing
August 2020

Ectopic cervical thymus in children: Clinical and radiographic features.

Laryngoscope 2020 06 28;130(6):1577-1582. Epub 2019 Aug 28.

Department of Otolaryngology, Head & Neck Surgery, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.

Objectives: Ectopic thymus is rare and can be a diagnostic challenge. This study evaluated the management of children radiographically diagnosed with ectopic cervical thymus.

Methods: A retrospective review of 100 patients was performed. Data related to clinical presentation, radiological imaging, pathology, and management were collected. Changes in lesion volume were tracked over time. Clinical characteristics were compared based on lesion location in the neck using analysis of variance modelling.

Results: There were 115 lesions with radiographic features of ectopic cervical thymus (15 children had bilateral lesions). Diagnosis was based on ultrasound in 98% of patients, magnetic resonance imaging in 18%, and computed tomography in 11%. Mean (SD) follow-up duration was 2 (2.2) years. Forty-four percent (51/115) of lesions involved the thyroid gland, 29% (33/115) were in the central neck but separate from the thyroid, 18% (21/115) had mediastinal extension, and 8% (9/115) involved the submandibular region. Location was unclear for two patients. Submandibular lesions were on average 12.4 cm larger (95% CI, 8.2, 16.6) than mediastinal lesions at diagnosis, P ≤ .001. Volume of thymic tissue decreased over time, from a mean (standard deviation [SD]) volume of 4.3 cm (9.2) at initial ultrasound to 2.7 cm (6.1) at final ultrasound (paired t-test, P = .008). Only two patients required surgery: one for compressive symptoms, and the other to rule out malignancy.

Conclusion: Ninety-eight percent of children with ectopic cervical thymus were managed conservatively without issues. We propose a classification system based on location to ease communication among clinicians and to help follow these lesions over time.

Level Of Evidence: 4, case series Laryngoscope, 130:1577-1582, 2020.
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http://dx.doi.org/10.1002/lary.28248DOI Listing
June 2020

Atypical juvenile histiocytosis with novel KIF5B-ALK gene fusion mimicking subglottic hemangioma.

Int J Pediatr Otorhinolaryngol 2019 Nov 13;126:109585. Epub 2019 Jul 13.

Department of Otolaryngology, Hospital for Sick Children, Toronto, ON, Canada; Department of Otolaryngology, Head and Neck Surgery, Faculty of Medicine, University of Toronto, Toronto, ON, Canada. Electronic address:

Juvenile Xanthograuloma (JXG) is part of a diverse set of rare histiocytic disorders marked by infiltration of tissues with neoplastic myelomonocytic-derived cells. Molecular analysis has yielded new insights into the classification and management of histiocytic diseases. A three-year-old presented with atypical croup due to a localized subglottic histiocytic lesion mimicking subglottic hemangioma. The lesion was removed via tracheofissure. Pathology revealed a JXG-like histopathology with a rare KIF5B-ALK fusion gene. This is the first isolated ALK-positive lesion to be reported in this location. The discovery of the new ALK-positive subclass of histiocytosis has opened the door for targeted monoclonal ALK inhibition.
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http://dx.doi.org/10.1016/j.ijporl.2019.07.010DOI Listing
November 2019

Pediatric nasopharyngeal airways expand when exposed to saline.

Int J Pediatr Otorhinolaryngol 2019 Nov 23;126:109595. Epub 2019 Jul 23.

Department of Otolaryngology-Head and Neck Surgery, Hospital for Sick Children, University of Toronto, ON, Canada. Electronic address:

Purpose: Nasopharyngeal airways (NPA) are commonly used to relieve upper airway obstruction in children. They must be sized to extend posterior to the tongue base but remain above the epiglottis. To avoid obstruction from nasal secretions, frequent irrigation with saline is required. We hypothesized that NPAs would swell when exposed to saline irrigation.

Methods: Twenty-five green Rusch NPA size 12, 14, 16, 18 and 20 Fr were submerged in 2 L of normal saline for 15 days. Tube length, inner diameter, outer diameter and wall thickness were measured on days 1,2,3,4,5,10, and 15 and compared using two-way repeated measures ANOVA and paired t-tests with Bonferroni correction.

Results: All dimensions increased significantly with exposure to saline. Increases in length, inner diameter, outer diameter and wall thickness were dependent on original tube size (p < 0.05) and duration of exposure to saline (p < 0.001). The increase in tube length was greatest over the first five days.

Conclusion: NPAs expand significantly when exposed to saline with the greatest increase in length occurring in the first five days. This could lead to gagging or airway obstruction in small children. Patients with NPAs should be monitored closely for these signs and new materials should be sourced to prevent these issues.
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http://dx.doi.org/10.1016/j.ijporl.2019.109595DOI Listing
November 2019

Obstructive sleep apnea in children aged 3 years and younger: Rate and risk factors.

Paediatr Child Health 2020 Nov 10;25(7):432-438. Epub 2019 Jul 10.

Division of Respiratory Medicine, Hospital for Sick Children, Toronto, Ontario.

Objective: Undiagnosed and untreated obstructive sleep apnea (OSA) can predispose children to neurobehavioural consequences. However, there is a lack of data identifying rate of, and risk factors for, OSA in very young healthy children. The objective of this study was to determine the rate of OSA and identify risk factors associated with the presence and severity of OSA in children aged 3 years and younger.

Methods: This was a retrospective chart review of healthy children between 1 and 3 years old who had a baseline polysomnogram (PSG) between January 2012 and June 2017. Patient demographics, referral history, and PSG data were recorded.

Results: One hundred and thirteen children were referred for a PSG, of which 66 (58%) were diagnosed with OSA and 47 (42%) did not have OSA. In the OSA group, 13 (20%) were mild and 53 (80%) were moderate-severe. Nasal congestion (P=0.001), adenoid hypertrophy (P=<0.001), and tonsillar hypertrophy (P=0.04) reported at the time of referral were more common in the OSA group compared to the no-OSA group. Binary logistic regression analysis showed that referral from an otolaryngologist (odds ratio=2.6, 95% confidence interval=1.1 to 6.0) were associated with moderate-severe OSA.

Conclusion: A high rate of OSA was found among children aged 3 years and younger. Children referred by an otolaryngologist are more likely to be diagnosed with moderate-severe OSA. Children aged 3 years and younger with symptoms of OSA should be considered high-risk for OSA and be prioritized for early PSG and management.
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http://dx.doi.org/10.1093/pch/pxz097DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7606157PMC
November 2020

Contact sport related head and neck injuries in pediatric athletes.

Int J Pediatr Otorhinolaryngol 2019 Jun 27;121:6-9. Epub 2019 Feb 27.

Department of Otolaryngology - Head and Neck Surgery, Faculty of Medicine, University of Toronto, Toronto, ON, Canada; Division of Otolaryngology - Head & Neck Surgery, Trillium Health Partners, Mississauga, ON, Canada. Electronic address:

Objectives: To describe the type and frequency of sports related pediatric head and neck trauma.

Methods: The National Electronic Injury Surveillance System (NEISS) was searched for football, basketball, soccer, lacrosse and ice hockey related facial injuries. Cross-sectional analysis of incidence, age, and sex and specific injury diagnoses, mechanisms, and facial locations were performed. We focused on craniofacial and soft tissue injuries and excluded patients with concussion or other neurological injuries.

Results: A total of 24,905 cases were identified in the NEISS database, corresponding to an estimated, 764,293 emergency department visits. The most hazardous sports were basketball, accounting for 356,188 visits (46.6%), football with 249,633 visits (32.6%), and soccer with 128,113 (14.7%) visits. Lacrosse and ice hockey accounted for 16,869 (1.9%) and 13,490 (1.5%) visits, respectively. There has been a decrease in injuries over the past 10 years, particularly for football (53%), ice hockey (46%), and lacrosse (41%). Soccer (13%) and basketball (9%) noticed the smallest decreases.

Conclusion: Children who play basketball, football and soccer are especially prone to emergency department visits related to the head and neck. Overall, there has been an improvement in number of injuries across the five sports investigated. Sports amenable to head and neck protective equipment saw the largest improvement (football, ice hockey, lacrosse), whereas only small decreases were noted in basketball and soccer. Changes in regulations for sports not amenable to more protective equipment may help decrease adverse events.
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June 2019

Surgery for Obstructive Sleep Apnea in Obese Children: Literature Review and Meta-analysis.

Otolaryngol Head Neck Surg 2019 06 19;160(6):985-992. Epub 2019 Feb 19.

1 Department of Otolaryngology-Head and Neck Surgery, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.

Objectives: Surgical intervention for obstructive sleep apnea (OSA) in overweight and obese children may not be as effective as it is in normal-weight children. The purpose of this study was to systematically review the effects of various surgical interventions for OSA in obese children and to meta-analyze the current data.

Data Sources: PubMed, OVID, and Cochrane databases.

Review Methods: Databases were searched for studies examining adenotonsillectomy, uvulopalatopharyngoplasty, supraglottoplasty, or tongue base surgeries and combinations in obese children with OSA. Adenotonsillectomy was the only procedure with enough data for meta-analysis; polysomnographic data were extracted and analyzed using a random-effects model.

Results: For adenotonsillectomy, 11 studies were included in the meta-analysis. Despite significant improvement in the apnea-hypopnea index (22.9 to 8.1 events/h, P < .001), respiratory disturbance index (24.8 to 10.4 events/h, P < .001), and oxygen saturation nadir (78.4% to 87.0%, P < .001), rates of persistent OSA ranged from 51% to 66%, depending on the outcome criterion used. There was evidence of limited effectiveness for surgical interventions to treat OSA in obese children using uvulopalatoplasty (12.5%) and tongue base surgery (74%-88%).

Conclusions: Surgical interventions for OSA in overweight and obese children are effective at reducing OSA but with higher rates of persistent OSA than reported for normal-weight children. However, the amount of reduction appears to vary by surgical procedure. More attention should be paid toward preoperative weight loss and patient selection, and parents should be provided with realistic postoperative expectations in this difficult-to-treat population.
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http://dx.doi.org/10.1177/0194599819829415DOI Listing
June 2019

Tracheo-innominate fistula in children: A systematic review of literature.

Laryngoscope 2020 01 10;130(1):217-224. Epub 2019 Jan 10.

Department of Otolaryngology-Head & Neck Surgery, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.

Objective: Tracheo-innominate fistula (TIF) is a rare but fatal complication of tracheotomy. To date, there is a paucity of literature regarding pediatric TIFs. The objectives of this study were to conduct a systematic review of literature on pediatric TIF following tracheotomy and describe three demonstrative cases from our institutional experience.

Methods: We conducted a systematic review using MEDLINE, Embase, Cochrane Database of Systematic Reviews, Web of Science, and CINAHL. All studies with pediatric patients (under 18 years of age) who developed TIF following tracheotomy were included.

Results: Fifty-four publications met inclusion criteria, reporting on 77 cases. The most common indication for tracheotomy was prolonged intubation and the need for ventilatory support (38.6%), with neurological comorbidities being the most common indication (72.7%). The mean time to TIF was 395.7 days (95% confidence interval, 225.9-565.5). Fifty-four patients (70.1%) presented with massive hemorrhage, whereas 18 patients (23.3%) presented with a sentinel bleeding event. The most common diagnostic interventions were computed tomography scan with or without contrast and bronchoscopy (55.8%). A substantial number of patients did not have any investigations (41.6%). Surgical management occurred in 70.1% of patients. Mortality was 38.6% in reported cases with variable follow-up periods.

Conclusion: TIF may occur in long-term tracheostomy-dependent children, contrary to the conventionally described 3-week postoperative period. The mortality may not be as high as previously reported with timely intervention. Our results are limited by inherent risks of bias. Further research including well-designed cohort studies are needed to guide an evidence-based approach to TIF.

Level Of Evidence: NA Laryngoscope, 130:217-224, 2020.
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http://dx.doi.org/10.1002/lary.27765DOI Listing
January 2020

A tyrosine sulfation-dependent HLA-I modification identifies memory B cells and plasma cells.

Sci Adv 2018 11 7;4(11):eaar7653. Epub 2018 Nov 7.

Department of Immunology, University of Toronto, Toronto, ON, Canada.

Memory B cells and plasma cells are antigen-experienced cells tasked with the maintenance of humoral protection. Despite these prominent functions, definitive cell surface markers have not been identified for these cells. We report here the isolation and characterization of the monoclonal variable lymphocyte receptor B (VLRB) N8 antibody from the evolutionarily distant sea lamprey that specifically recognizes memory B cells and plasma cells in humans. Unexpectedly, we determined that VLRB N8 recognizes the human leukocyte antigen-I (HLA-I) antigen in a tyrosine sulfation-dependent manner. Furthermore, we observed increased binding of VLRB N8 to memory B cells in individuals with autoimmune disorders multiple sclerosis and systemic lupus erythematosus. Our study indicates that lamprey VLR antibodies uniquely recognize a memory B cell- and plasma cell-specific posttranslational modification of HLA-I, the expression of which is up-regulated during B cell activation.
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http://dx.doi.org/10.1126/sciadv.aar7653DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6221509PMC
November 2018

Tracheostomy care: Clinical practice patterns of pediatric otolaryngologists-head and neck surgeons in a publicly funded (Canadian) health care system.

Int J Pediatr Otorhinolaryngol 2018 Dec 25;115:177-180. Epub 2018 Sep 25.

Division of Respiratory Medicine, Hospital for Sick Children, Department of Pediatrics, University of Toronto, Canada. Electronic address:

Objectives: To investigate variability in pediatric tracheostomy tube care practice patterns and access to resources across Canada.

Methods: Canadian pediatric otolaryngologists-head & neck surgeons reported their own practice patterns for children with chronic tracheostomy tubes using a web-based, 29-item multiple choice and short answer questionnaire. Domains investigated included tracheostomy team membership, inpatient care practices, caregiver education, homecare resources, speech and communication, and completeness of emergency tracheostomy kits.

Results: The response rate was 86.4% (38/44). Most respondents care for children with tracheostomy tubes as part of an inter-professional team (25/36; 69.4%) and arrange routine follow-up with a speech and language pathologist (22/36; 61.1%). However, the majority (23/34; 67.6%) of respondents do not formally reassess caregiver competencies (i.e. cardiopulmonary resuscitation, emergency tracheostomy care). Notably, respondents were also unsure 36.1% (13/36) of how frequently Shiley tracheostomy tubes should be washed and reused with the majority (15/36; 41.7%) reporting never. Most (15/36; 41.7%) respondents were also unsure of reuse recommendations for Bivona tracheostomy tubes. One third (12/36; 33.3%) of respondents were unsure about government-funded homecare services being provided in their community to children with tracheostomy tubes.

Conclusion: There is much variability in pediatric tracheostomy tube care practice patterns across Canada. Results suggest that an evidence-based Canadian clinical practice guideline may help to streamline care provided to Canadian children with tracheostomy tubes.
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http://dx.doi.org/10.1016/j.ijporl.2018.09.020DOI Listing
December 2018

Airway Compression After Unifocalization in Pulmonary Atresia and Aortopulmonary Collateral Arteries.

Ann Thorac Surg 2019 03 23;107(3):844-851. Epub 2018 Oct 23.

Division of Cardiovascular Surgery, The Labatt Family Heart Centre, The Hospital for Sick Children, Toronto, Ontario, Canada; Department of Surgery, University of Toronto, Toronto, Ontario, Canada. Electronic address:

Background: We hypothesized that reconstructed pulmonary artery (PA) size and postrepair PA pressure are associated with airway compression (AC) after complete unifocalization for pulmonary atresia, ventricular septal defects, and major aortopulmonary collateral arteries.

Methods: Complete unifocalization was performed in 48 consecutive patients between 2000 and 2016. Clinical course and outcome were reviewed, predictors for AC were identified by logistic regression, and the freedom from death was analyzed using Kaplan-Meier method.

Results: Postoperative respiratory distress occurred in 23 patients (48%), and AC occurred in 14 (29%). The median duration of follow-up was 3.7 years. AC was caused by central PA and aorta in 7, conduit in 3, and branch PA in 4. Surgical treatment was required in 5 patients (conduit downsizing, suspension of branch PA, conduit + aorta, branch PA + aorta, and aorta + trachea in 1 patient each). Three patients (21%) subsequently required airway stenting. Most (85.7%) of the AC occurred in patients with high right ventricular systolic pressure/left ventricular systolic pressure (>65%), large Nakata index (>200 mm/m), and large conduit index (>35 mm/m). Patients with AC had significantly worse 3-year survival (no AC, 91.2%; AC, 64.2%; p = 0.01). Multivariate analysis identified higher right ventricular systolic pressure/left ventricular systolic pressure (p = 0.04), larger conduit index (p = 0.03), and Nakata index (p = 0.004) as predictors for AC.

Conclusions: AC is a common cause of postoperative respiratory distress and tends to be associated with higher postrepair PA pressure, more frequent right ventricular dysfunction, and worse medium-term survival. The study underscores the importance of incorporating all available lung segments to achieve a low PA pressure, potentially preventing pathologic dilatation of the reconstructed PA. Management of patients with poor major aortopulmonary collateral arteries anatomy and physiology remains a challenge.
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http://dx.doi.org/10.1016/j.athoracsur.2018.08.090DOI Listing
March 2019

Children with unilateral cochlear nerve canal stenosis have bilateral cochleovestibular anomalies.

Laryngoscope 2019 10 23;129(10):2403-2408. Epub 2018 Oct 23.

Department of Otolaryngology-Head and Neck Surgery, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.

Objectives/hypothesis: To investigate the cochleovestibular apparatus bilaterally in children with isolated unilateral bony cochlear nerve canal (bCNC) stenosis.

Study Design: Retrospective review.

Methods: Imaging studies of children with unilateral bCNC stenosis (<1.0 mm) on computed tomography imaging (N = 36) were compared with controls imaged due to trauma without temporal bone injury (N = 32). Twenty-six measurements were obtained in each ear, assessing the bony internal auditory canal (IAC), cochlea, and vestibular end-organs, and were analyzed using one-way analysis of variance for intersubject comparisons and paired t tests for intrasubject comparisons with a Bonferroni adjustment for multiple comparisons (P = .0006).

Results: Patients with bCNC stenosis had a smaller IAC (P < .000) and cochlea (P < .000) on the stenotic side as compared with controls. Although the vestibular end-organ was also smaller in bCNC ears, this difference was not significant. The contralateral ear also had a smaller bCNC (P < .000) and cochlea (P < .000) as compared with controls, although to a lesser degree than the stenotic side.

Conclusions: Children with unilateral bCNC stenosis have abnormal biometry of both the cochlea and the vestibular end-organ in the affected and the normal contralateral ear as compared with controls.

Level Of Evidence: 3b Laryngoscope, 129:2403-2408, 2019.
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http://dx.doi.org/10.1002/lary.27559DOI Listing
October 2019