Objective To identify the ability of optical coherence tomography (OCT) to assist in the diagnosis and treatment of patients presenting to a primary care provider with signs and symptoms suggesting acute otitis media (AOM). Design-Setting-Participants An NIH-funded clinical trial was conducted and enrolled 75 children between 1 and under 7 years of age. Children presented with complaints of ear pain or suspected ear infection from their caregiver history to primary care clinics associated with an academic children’s hospital. These patients upon presentation were eligible to enroll in the trial. Following a complete history and physical exam, including pneumatic otoscopy (PO), patients were provided with a diagnosis and treatment plan. Diagnoses included: AOM, middle ear effusion without AOM, or no evidence of otitis media with a clear middle ear space. Following this, OCT was performed, and the diagnosis and treatment plan was reassessed using this data. Results The diagnosis and/or treatment plan changed for 15.3% of patients following the use of OCT compared with the initial use of PO. The greatest influence was seen in the patient group with MEE without AOM, with 36% (5/14) of these patients having their diagnosis and/or treatment plan changed after OCT was employed. Conclusions OCT provides an additional adjunct for primary care providers to enhance visualization of the tympanic membrane and middle ear space. This data, in our clinical trial, resulted in enhanced clinical decision making, in particular for patients with MEE who were not diagnosed with AOM on PO. Trial Registration Information Coherent Optical Detection of Middle Ear Disease (OCTII). Clinicaltrials.gov ID number: NCT05353569

INTRODUCTION:Pediatric head and neck lymphatic malformations (HNLM) are heterogeneous lesions that often require multimodal care. Contemporary real-world data incorporating evolving interventional techniques and targeted systemic therapy are limited. We describe patient characteristics, imaging utilization, treatment utilization/sequencing, and 90-day treatment-related complications within a multidisciplinary vascular anomalies program. METHODS:Retrospective single-institution chart review of patients <18 years with head and neck lymphatic malformations through the Birthmarks and Vascular Anomalies Program (BVAP) from 2014 to 2024 (n = 59). Demographics, lesion characteristics, imaging, treatment sequencing, and 90-day treatment-related complications were abstracted from the medical record. Analyses were descriptive; procedure-level complication rates were reported with exact 95% confidence intervals (CIs). RESULTS:Morphology was 42.4% microcystic, 37.3% macrocystic, and 20.3% mixed; 39.0% were multiregional. Median lesion diameter was 4.0 cm (IQR 2.1-6.4). MRI was obtained in 93.2% of patients. Sclerotherapy was used in 67.8% (86 sessions), most commonly doxycycline (45.8%) and bleomycin (35.6%). Resection without embolization occurred in 45.8%, laser therapy in 15.3%, and glue embolization surgery in 5.1%. Systemic therapy included sirolimus in 25.4%, with 4 patients transitioning to alpelisib. Eight patients (13.6%) underwent molecular testing; all tested had pathogenic PIK3CA variants. Ten patients (16.9%) experienced ≥1 complication. Procedure-level complications occurred after 5/86 sclerotherapy procedures (5.8%; 95% CI 1.9-13.0), 6/53 resections without embolization (11.3%; 95% CI 4.3-23.0), and 0/3 glue embolization procedures (0.0%; 95% CI 0.0-70.8). CONCLUSION:Pediatric HNLM often required multimodal, staged treatment with high MRI utilization and repeat procedures. Prospective multicenter studies with standardized outcomes and systematic molecular testing are needed to guide lesion-specific care.

PURPOSE/OBJECTIVE:To investigate the utility of Sinus CT imaging in the diagnosis or suspicion of invasive fungal sinusitis in immunocompromised children. STUDY DESIGN:Retrospective chart review. SETTING:Tertiary-care pediatric hospital. SUBJECTS AND RESULTS:Between 2012 and 2020, the Otolaryngology service at Children's Wisconsin was consulted 102 separate times in 83 patients for concern for persistent fevers of unknown origin in immunocompromised patients with concern for invasive fungal infection (IFS). There was a CT scan associated with consultation in 82 (80 %) and 49 % had been obtained prior to Otolaryngology consultation. IFS was confirmed on biopsy on 8 (10 %) of patients and 6 (7 %) had concerning findings on bedside nasal endoscopy. The remaining two patients had congestion but lacked other findings on bedside nasal endoscopy. However, they were taken to the OR for facial swelling (1/8) for high clinical suspicion prompting a CT which demonstrated a septal abscess. CONCLUSIONS:CT imaging is a highly used modality for the evaluation of invasive fungal sinusitis. Our data demonstrates no significant difference between the CTs ordered on patients with invasive fungal disease and those without. Bedside nasal endoscopy remained the primary driver for operative biopsy, though, one case was prompted by CT imaging. We advocate for bedside nasal endoscopy as the primary screening in these patients. However, for those with a negative bedside nasal exam but a high index of suspicion, CT imaging may play a role in further evaluation.

BACKGROUND:Tympanostomy tube (TT) insertion is typically performed in children in the operating room under general anesthesia. Devices combining equipment and steps in TT insertion, facilitate TT placement and may enable a transition to in-office TT procedures avoiding general anesthesia. This study evaluates the safety and effectiveness of a new TT insertion device, Solo + TTD, with general anesthesia. METHODS:Children aged 6 months to 15 years meeting criteria for TT insertion were enrolled in an IRB-approved multicenter single-arm prospective study. TT insertion was performed in the operating room under general anesthesia using the study device. Intraoperative device success, rate of adverse events, function of ear tubes during follow-up (1, 3, 6, 12, 18 and 24 months), and use of additional instrumentation for placement of Solo + TT were evaluated. Interim data through 12 months of follow up are presented in this paper. RESULTS:A total of 59 ears in 30 patients were treated at two sites. The mean age of enrolled children was 2.4 years and 57 % were male. Solo + TT insertion was successful (± use of additional instrumentation) in 57/59 (96.6 %) ears with unsuccessful insertion in two patients due to irregular ear canal anatomy. Two ears required a second Solo + TTD cartridge for successful insertion. There were no serious adverse events related to the device or procedure. Non-serious adverse events at one-month (most common bleeding, otorrhea and blockage) occurred at rates (9/30 patients) similar to standard TT. At 1-month follow up, 96 % (51/53) ears had functioning TT. Through 12-months follow-up, 20 % of TT were extruded (9/44 ears) and 88.6 % of present TT (31/35 ears) were functioning. CONCLUSION:The Solo + TTD tympanostomy tube device has a high rate of successful TT placement in children under general anesthesia. The device TTs function is similar to traditional TTs and other device TTs out to 1 year. Additional studies evaluating the device in children using topical anesthesia are needed.

Objective: To provide recommendations for a comprehensive management approach for infants and children presenting with symptoms or signs of aspiration. Methods: Three rounds of surveys were sent to authors from 23 institutions worldwide. The threshold for the critical level of agreement among respondents was set at 80 %. To develop the definition of "intractable aspiration," each author was first asked to define the condition. Second, each author was asked to complete a 5-point Likert scale to specify the level of agreement with the definition derived in the first step. Results: Recommendations by the authors regarding the clinical presentation, diagnostic considerations, and medical and surgical management options for aspiration in children. Conclusion: Approach to pediatric aspiration is best achieved by implementing a multidisciplinary approach with a comprehensive investigation strategy and different treatment options.

Introduction: Non-tuberculous mycobacterial (NTM) infection commonly manifests as subacute or chronic cer-vicofacial lymphadenitis in immunocompetent children. The optimal management of this pathology remains controversial. Objectives: This international consensus guideline aims to understand the practice patterns for NTM cervicofacial lymphadenitis and to address the primary diagnostic and management challenges.Methods: A modified three-iterative Delphi method was used to establish expert recommendations on the diag-nostic considerations, expectant or medical management, and operative considerations. The recommendations herein are derived from current expert consensus and critical review of the literature. Setting: Multinational, multi-institutional, tertiary pediatric hospitals. Results: Consensus recommendations include diagnostic work-up, goals of treatment and management options including surgery, prolonged antibiotic therapy and observation. Conclusion: The recommendations formulated in this International Pediatric Otolaryngology Group (IPOG) consensus statement on the diagnosis and management of patients with NTM lymphadenitis are aimed at improving patient care and promoting future hypothesis generation.

Objective: Examine differences in cost between single stage (ss) versus double stage (ds) laryngotracheal reconstruction (LTR) for pediatric subglottic stenosis. Study design: Retrospective chart review of children who underwent ssLTR or dsLTR from 2014 to 2018 at a single institution.Methods: Costs related to LTR and post-operative care up to one year after tracheostomy decannulation were extrapolated from charges billed to the patient. Charges were obtained from the hospital finance department and the local medical supplies company. Patient demographics including baseline severity of subglottic stenosis and co-morbidities were noted. Variables assessed include duration of hospital admission, number of ancillary procedures, duration of sedation wean, cost of tracheostomy maintenance, and time to tracheostomy decannulation.Results: Fifteen children underwent LTR for subglottic stenosis. D Ten patients underwent ssLTR, while five underwent dsLTR. Grade 3 subglottic stenosis was more prevalent in patients who underwent dsLTR (100%) than ssLTR (50%). The average per-patient hospital charges for ssLTR was $314,383 versus $183,638 for dsLTR. When estimated mean cost of tracheostomy supplies and nursing care until tracheostomy decannulation was included, the mean total charges associated with dsLTR patients was $269,456. Average hospital stay after initial surgery was 22 days for ssLTR versus 6 days for dsLTR. Average time to tracheostomy decannulation for dsLTR was 297 days. Average number of ancillary procedures needed was 3 versus 8 for ssLTR versus dsLTR.Conclusion: For pediatric patients with subglottic stenosis, dsLTR may have a lower cost than ssLTR. Although ssLTR has the benefit of immediate decannulation, it is associated with higher patient charges, as well as longer initial hospitalization and sedation. For both patient groups, fees associated with nursing care comprised the majority of charges. Recognizing the factors that contribute to cost differences between ssLTR and dsLTR may be useful when performing cost-benefit analyses and assessing value in health care delivery.

Adenotonsillectomy is the first-line treatment for pediatric obstructive sleep apnea (OSA). The postoperative course may be complicated by hypoxia, requiring intervention. Positive pressure respiratory support (PPS) could be used to bridge the postoperative period and avoid invasive mechanical ventilation; however, the safety of PPS following tonsillectomy has not been established. Objective To review the incidence of complications and risk factors associated with PPS use immediately after tonsillectomy.A retrospective cohort study between 2015 and 2020 of patients who underwent tonsillectomy and were admitted to the pediatric intensive care unit at a single healthcare system.Seven hundred eighty patients met inclusion criteria, including 101 patients treated with PPS immediately following surgery. A similar number of patients were diagnosed with severe OSA in each group prior to surgery. One patient in the PPS cohort developed pneumomediastinum and pneumothorax. Eleven patients (12%) in the PPS group and 18 patients (2%) in the non-PPS group developed life-threatening complications, defined as pneumothorax/pneumomediastinum, re-intubation, post-tonsillectomy bleeding that required surgical intervention, pulmonary edema and death, and all occurred in patients who had not used PPS at baseline. Regression analysis identified body mass index, surgical technique, and PPS use to be associated with increased odds of life-threatening complications.Our study suggests that PPS is generally safe to use. New-onset PPS is associated with increased odds of life-threatening complications, likely reflecting a severe post-surgical clinical course.

OBJECTIVE:Lymphatic malformations in the submandibular neck pose unique challenges to treatment that elevate their risk of recurrence. This case series provides a review of five patients, previously treated with sclerotherapy or with a history of multiple infections, who were treated in a novel fashion: single-stage resection using preoperative n-butyl cyanoacrylate (n-BCA) glue embolization.METHODS:We performed a retrospective medical record review of five patients who underwent single-stage n-BCA embolization by Interventional Radiology followed by surgical resection by Otolaryngology, including a review of their symptoms, previous treatments, and post-treatment surveillance, with follow-up ranging from 4 to 24 months after the treatment of interest.RESULTS:All study subjects had unremarkable perioperative courses, and four patients did not demonstrate any evidence of disease recurrence or persistence during the follow-up period. One patient was found to have a small area of persistent disease on post-treatment imaging, but has remained symptom free.CONCLUSIONS:Treatment of submandibular lymphatic malformations with n-BCA embolization followed by surgical resection can be performed in a single stage. This case series demonstrates that this approach can yield durable relief of symptoms, even in patients whose lesions were refractory to previous treatments.

Objective: Otitis media (OM) is among the most frequently diagnosed pediatric diseases in the US. Despite the significant public health burden of OM and the contribution research in culture models has made to understanding its pathobiology, a singular immortalized human middle ear epithelial (MEE) cell line exists (HMEEC-1, adult-derived). We previously developed MEE cultures from pediatric patients with non-inflamed MEE (PCI), recurrent OM (ROM), or OM with effusion (OME) and demonstrated differences in their baseline inflammatory cytokine expression and response to stimulation with an OM-relevant pathogen lysate and cytokines. Herein, we sought to immortalize these cultures and assess retention of their phenotypes.Methods: MEE cultures were immortalized via lentivirus encoding temperature-sensitive SV40 T antigen. Immortalized MEE lines and HMEEC-1 grown in monolayer were stimulated with non-typeable Haemophilus influenzae (NTHi) lysate. Gene expression (TNFA, IL1B, IL6, IL8, MUC5AC, and MUC5B) was assessed by qPCR.Results: Similar to parental cultures, baseline cytokine expressions were higher in pediatric OM lines than in HMEEC-1 and PCI, and HMEEC-1 cells were less responsive to stimulation than pediatric lines.Conclusion: Immortalized MEE lines retained the inflammatory expression and responsiveness of their tissues of origin and differences between non-OM versus OM and pediatric versus adult cultures, supporting their value as novel in vitro culture models for OM.

Management of tongue venous malformations can be challenging in the pediatric population due to their heterogeneity in presentation, extent of involvement and functional compromise. It is important to recognize the value of various treatment options in order to guide management of each patient in an individualized fashion. Here we describe a series of patients with tongue venous malformations that are managed using diverse modalities to illustrate the relative benefits and risks of each technique. The challenges of venous malformation treatment can be mitigated by tailoring the approach to each individual patient and malformation. This case series also emphasizes the need and importance of working in the setting of a multidisciplinary vascular anomalies team.

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.

ObjectiveOtitis media (OM) is a common inflammatory disease spectrum. Cytokine signaling, neutrophil activity, and mucin hypersecretion during recurrent and chronic OM contribute to persistent, viscous middle ear (ME) effusions, hearing loss, and potential for developmental delay. Extraesophageal reflux (EER), specifically pepsin, triggers inflammatory signaling in respiratory mucosa and is associated with OM. The objective of this study was to investigate the association of pepsin with ME inflammatory signaling and the outcomes and examine causality in vitro.Study DesignCross‐sectional study.MethodsME fluid (MEF) and preoperative audiometric data were collected from 30 pediatric subjects undergoing tympanostomy tube placement for recurrent OM or OM with effusion. MEF viscosity was characterized by the surgeon. Pepsin, inflammatory molecules, and mucin were assayed by enzyme‐linked immunosorbent assay (ELISA). ME epithelial primary culture was exposed to 0.1 to 1 mg/ml pepsin at pH 5, 6, and 7 for 30 minutes, and cytokine expression was assayed via qPCR.ResultsPepsin was observed in the MEF of 77% of patients (range 71–2,734 ng/ml). Pepsin correlated with effusion viscosity, interleukins −6 and −8, neutrophil elastase, and mucin 5B (P < .05). Pepsin‐negative MEF was more frequently absent of interleukin 8 or mucin 5B (P < .05). Weak acid was generally insufficient to elicit cytokine expression in ME cells in vitro, however, pepsin induced IL6, IL8, and TNF at pH 7 (P < .05) and weak acid (pH 6) facilitated a response at lower pepsin concentration.ConclusionsPepsin may contribute to inflammatory signaling, persistent viscous effusion, and poorer OM outcomes.Level of Evidence4 Laryngoscope, 132:470–477, 2022

OBJECTIVES:Otitis media (OM) is the most common pediatric diagnosis in the United States. However, our understanding of the molecular pathogenesis of OM remains relatively poor. Investigation of molecular pathways involved in OM may improve the understanding of this disease process and elucidate novel therapeutic targets. In this study, RNA sequencing (RNA-Seq) was used to discern cellular changes associated with OME compared to healthy middle ear epithelium (MEE). STUDY DESIGN:Ex vivo case-control translational. METHODS:Middle ear epithelia was collected from five pediatric patients diagnosed with OME undergoing tympanostomy tube placement and five otherwise healthy pediatric patients undergoing cochlear implantation. Specimens underwent RNA-Seq and pathways analyses. RESULTS:A total of 1,292 genes exhibited differential expression in MEE from OME patients compared to controls including genes involved in inflammation, immune response to bacterial OM pathogens, mucociliary clearance, regulation of proliferation and transformation, and auditory cell differentiation. Top networks identified in OME were organismal injury and abnormalities, cell morphology, and auditory disease. Top Ingenuity canonical pathways identified were axonal guidance signaling, which contains genes associated with auditory development and disease and nicotine degradation II and III pathways. Associated upstream regulators included β-estradiol, dexamethasone, and G-protein-coupled estrogen receptor-1 (GPER1), which are associated with otoprotection or inflammation during insult. CONCLUSIONS:RNA-Seq demonstrates differential gene expression in MEE from patients with OME compared to healthy controls with important implications for infection susceptibility, hearing loss, and a role for tobacco exposure in the development and/or severity of OME in pediatric patients. LEVEL OF EVIDENCE:4 Laryngoscope, 131:2590-2597, 2021.

Objectives: To provide recommendations to otolaryngologists, pulmonologists, and allied clinicians for tracheostomy decannulation in pediatric patients. Methods: An iterative questionnaire was used to establish expert recommendations by the members of the International Pediatric Otolaryngology Group. Results: Twenty-six members completed the survey. Recommendations address patient criteria for decannulation readiness, airway evaluation prior to decannulation, decannulation protocol, and follow-up after both successful and failed decannulation. Conclusion: Tracheostomy decannulation recommendations are aimed at improving patient-centered care, quality and safety in children with tracheostomies.

Objectives/HypothesisCell culture models are valuable tools for investigation of the molecular pathogenesis of diseases including otitis media (OM). Previous study indicates that age‐, sex‐, and race‐associated differences in molecular signaling may impact disease pathophysiology. Currently, a singular immortalized middle ear epithelial (MEE) cell line exists, HMEEC‐1, derived from an adult without known middle ear disease. In this study, HMEEC‐1 and primary MEE cultures from pediatric patients with and without OM were stimulated with inflammatory cytokines or OM‐pathogenic bacterial lysates to examine differences in the response of molecules associated with OM pathogenesis.Study DesignCase–control series.MethodsMEE cultures were established from patients aged <6 years: two with recurrent OM (ROM), two with OM with effusion (OME), and one patient without OM who was undergoing cochlear implant surgery control undergoing cochlear implantation (Peds CI). Primary MEE cultures and HMEEC‐1 cells were stimulated with tumor necrosis factor‐α, interleukin (IL)‐1β, or nontypeable Haemophilus influenzae lysate. TNFA, IL1B, IL6, IL8, IL10, and MUC5B were assayed via quantitative polymerase chain reaction. IL‐8 was assayed by enzyme‐linked immunosorbent assay.ResultsGene/protein target expressions were frequently higher in pediatric OM lines than in HMEEC‐1 and Peds CI. HMEEC‐1 cells were frequently less responsive to stimuli than all pediatric lines. OME lines were often more responsive than ROM lines.ConclusionsOM may be associated with specific molecular phenotypes that are retained in primary cell culture. Adult‐derived HMEEC‐1 cells differ significantly in baseline expression and response of OM‐associated molecules relative to pediatric MEE cells. Work is underway to immortalize pediatric OM MEE cultures as improved tools for the OM research community.Level of Evidence4 Laryngoscope, 131:410–416, 2021