ABSTRACT
OBJECTIVES: To determine rates and risk factors of pediatric otitis media (OM) using real-world electronic health record (PEDSnet) data from January 2009 through May 2021. STUDY DESIGN: Retrospective cohort study. SETTING: Seven pediatric academic health systems that participate in PEDSnet. METHODS: Children <6 months-old at time of first outpatient, Emergency Department, or inpatient visit were included and followed longitudinally. A time-to-event analysis was performed using a Cox proportional hazards model to estimate hazard ratios for OM incidence based on sociodemographic factors and specific health conditions. RESULTS: The PEDSnet cohort included 910,265 children, 54.3% male, mean age (months) 1.3 [standard deviation (SD) 1.6] and mean follow up (years) 4.3 (SD 3.2). By age 3 years, 39.6% of children had evidence of one OM episode. OM rates decreased following pneumococcal-13 vaccination (PCV-13) and the COVID-19 pandemic. Along with young age, non-Hispanic Black/African American or Hispanic race/ethnicity, public insurance, higher family income, and male sex had higher incidence rates. Health conditions that increased OM risk included cleft palate [adjusted hazard ratio (aHR) 4.0 [95% confidence interval (CI) 3.9-4.2], primary ciliary dyskinesia [aHR 2.5 (95% CI 1.8-3.5)], trisomy 21 [aHR 2.0 (95% CI 1.9-2.1)], atopic dermatitis [aHR 1.4 (95% CI 1.4-1.4)], and gastroesophageal reflux [aHR1.5 (95% CI 1.5-1.5)]. CONCLUSIONS: Approximately 20% of children by age 1 and 40% of children by age 3 years will have experienced an OM episode. OM rates decreased after PCV-13 and COVID-19. Children with abnormal ciliary function or craniofacial conditions, specifically cleft palate, carry the highest risk of OM.
Subject(s)
Cleft Palate , Otitis Media , Child , Humans , Male , Infant , Child, Preschool , Female , Retrospective Studies , Cleft Palate/complications , Pandemics , Otitis Media/etiology , Risk FactorsABSTRACT
Background: Otitis media (OM) is defined as middle ear (ME) inflammation that is usually due to infection. Globally, OM is a leading cause of hearing loss and is the most frequently diagnosed disease in young children. For OM, pediatric patients with Down syndrome (DS) demonstrate higher incidence rates, greater severity, and poorer outcomes. However, to date, no studies have investigated the bacterial profiles of children with DS and OM. Method: We aimed to determine if there are differences in composition of bacterial profiles or the relative abundance of individual taxa within the ME and nasopharyngeal (NP) microbiotas of pediatric OM patients with DS (n = 11) compared with those without DS (n = 84). We sequenced the 16S rRNA genes and analyzed the sequence data for diversity indices and relative abundance of individual taxa. Results: Individuals with DS demonstrated increased biodiversity in their ME and NP microbiotas. In children with OM, DS was associated with increased biodiversity and higher relative abundance of specific taxa in the ME. Conclusion: Our findings suggest that dysbioses in the NP of DS children contributes to their increased susceptibility to OM compared with controls. These findings suggest that DS influences regulation of the mucosal microbiota and contributes to OM pathology.
Subject(s)
Down Syndrome , Microbiota , Otitis Media , Child , Humans , Child, Preschool , RNA, Ribosomal, 16S/genetics , Down Syndrome/genetics , Otitis Media/genetics , Ear, Middle/microbiology , Ear, Middle/pathology , Microbiota/geneticsABSTRACT
Laryngotracheal stenosis, congenital or acquired, is a common cause of pediatric airway obstruction. Acquired subglottic stenosis frequently results from prolonged neonatal intubation. The clinical presentation of subglottic stenosis is variable, ranging from biphasic stridor and frequent upper respiratory infections to acute airway compromise. Optimal patient care requires clinical coordination within a multidisciplinary subspecialty team. Medical management includes optimizing respiratory status, gastroesophageal reflux, speech, feeding, nutrition therapies, and providing psychosocial support. If surgical intervention is required, the otolaryngologist, anesthesiologist, and perioperative team must collaborate closely to ensure successful operative outcomes. This narrative review of laryngotracheal stenosis will discuss the pathophysiology, clinical evaluation, medical management, and surgical interventions, and focus on the perioperative anesthetic considerations for children undergoing laryngotracheal reconstruction.
Subject(s)
Anesthesia , Laryngostenosis , Plastic Surgery Procedures , Tracheal Stenosis , Infant, Newborn , Child , Humans , Constriction, Pathologic/complications , Constriction, Pathologic/surgery , Treatment Outcome , Laryngostenosis/surgery , Laryngostenosis/etiology , Tracheal Stenosis/surgery , Tracheal Stenosis/complications , Anesthesia/adverse effects , Retrospective StudiesABSTRACT
OBJECTIVES: Croup is characterized by a barky cough, inspiratory stridor, hoarseness and varying degrees of respiratory distress. Acute croup episodes are often treated with oral, inhaled, or intravenous corticosteroids. Recurrent croup, defined as more than 2-3 episodes of acute croup in the same patient, can mimic asthma. We hypothesized that inhaled corticosteroids (ICS) given at the first sign of a respiratory viral prodrome can be a safe treatment to reduce the frequency of recurrent croup episodes in children without fixed airway lesions. METHODS: A retrospective chart review of patients being treated over an 18-month period was performed at a large tertiary care pediatric hospital following Institutional Review Board (IRB) approval. Patients under 21 years old referred to Pediatric Pulmonology, Otolaryngology, or Gastroenterology for recurrent croup were analyzed for their demographics, medical history, evaluation, treatment and clinical improvement. A Fisher's two-tailed exact test was used to compare the number of croup episodes before and after interventions. RESULTS: 124 patients were included in our analysis: 87 male and 34 female with a mean age of 54 months. Of these, 78 had >5 episodes of croup, 45 had 3-5, and 3 had 2 episodes prior to their first visit for recurrent croup. Operative direct laryngoscopy/bronchoscopy was performed in 35 patients (27.8%), with 60% showing a normal exam without fixed lesions. Ninety-two patients (74.2%) were treated with ICS, 24 were lost to follow up. Of the remaining 68 treated patients, 59 (86.7%) saw improvement with reduced severity and overall number of episodes of croup. Additionally, patients with >5 episodes of croup (47) as compared to <5 (12) were more likely to improve with ICS, (p = 0.003). There were no adverse reactions reported with ICS treatment. CONCLUSION: The novel initiation of ICS at the earliest sign of a viral upper respiratory infection shows promise as a safe preventative treatment to mitigate the frequency of recurrent croup episodes.
Subject(s)
Asthma , Croup , Child , Humans , Male , Female , Child, Preschool , Young Adult , Adult , Croup/diagnosis , Retrospective Studies , Adrenal Cortex Hormones/therapeutic use , Cough , Asthma/diagnosis , Asthma/drug therapyABSTRACT
OBJECTIVES: Burnout among healthcare workers is a public health crisis. Burnout is associated with elevated cynicism, emotional exhaustion, and low job satisfaction. Methods to combat burnout have been challenging to identify. Based on positive experiences of pediatric aerodigestive team members, we hypothesized that social support in multidisciplinary aerodigestive teams moderates the effects of burnout on job satisfaction. METHODS: Using a survey of the Aerodigestive Society, members of Aerodigestive teams (N = 119) completed demographics, the Maslach Burnout Inventory, and measures of job satisfaction, emotional, and instrumental social support. In addition to assessing relationships between components of burnout and job satisfaction, six tests were conducted using PROCESS to ascertain the degree to which social support moderated these relationships. RESULTS: Similar to US healthcare base rates, burnout scores in this sample suggest that a third-to-half felt Emotionally Exhausted and Burned Out from work "A few times a month"-to-"Every Day." Simultaneously, however, the majority in sample (60.6%) noted feeling that they "positively impact others' lives" with 33.3% endorsing "Every Day." Job satisfaction was strikingly high at 89%, with most reporting Aerodigestive team affiliation related to higher job satisfaction. Both Emotional and Instrumental social support moderated the effect of Cynicism and Emotional Exhaustion on Job Satisfaction, with higher Job Satisfaction scores in conditions of high support. CONCLUSIONS: These results support the hypothesis that social support from a multidisciplinary aerodigestive team moderates the effect of burnout in its team members. Further work is needed to understand if membership in other interprofessional healthcare teams can help combat the negative effects of burnout.
Subject(s)
Burnout, Professional , Job Satisfaction , Humans , Child , Burnout, Professional/epidemiology , Burnout, Professional/psychology , Social Support , Emotions , Surveys and QuestionnairesABSTRACT
We report a series of four patients with severe bronchopulmonary dysplasia (BPD) who underwent posterior tracheopexy for severe tracheomalacia (TM). While posterior tracheopexy is an established surgical treatment for TM associated with tracheoesophageal fistula, it has not been previously described in TM associated with BPD. There were no significant intraoperative or postoperative complications from the surgeries. Three of the four patients required tracheostomy and mechanical ventilation, which may reflect the degree of lung disease and other multisystem comorbidities in these patients. More investigation is needed to determine whether posterior tracheopexy is an effective surgical option for TM related to BPD.
Subject(s)
Bronchopulmonary Dysplasia , Tracheobronchomalacia , Tracheoesophageal Fistula , Tracheomalacia , Bronchopulmonary Dysplasia/complications , Bronchopulmonary Dysplasia/surgery , Humans , Infant , Infant, Newborn , Infant, Premature , Tracheobronchomalacia/complications , Tracheobronchomalacia/surgery , Tracheoesophageal Fistula/surgery , Tracheomalacia/complications , Tracheomalacia/surgeryABSTRACT
Posterior tracheopexy, performed most often for intrinsic tracheomalacia, is a procedure that is gaining in popularity and application. In this procedure, pledgeted sutures are placed between the trachea and a more fixed structure, in order to provide the airway with greater patency. Polytetrafluoroethylene pledgets are readily available but risk migration into adjacent structures over time. We present a case of a pledget migrating in to the airway and presenting as an obstructing lesion that has required multiple repeat interventions. Given that bioabsorbable materials are available, surgeons should consider alternative pledgets for these patients.
Subject(s)
Polytetrafluoroethylene , Tracheomalacia , Bronchoscopy , Humans , Sutures/adverse effects , Trachea/surgeryABSTRACT
Otitis media (OM) is common in young children and can cause hearing loss and speech, language, and developmental delays. OM has high heritability; however, little is known about OM-related molecular and genetic processes. CDHR3 was previously identified as a locus for OM susceptibility, but to date, studies have focused on how the CDHR3 p.Cys529Tyr variant increases epithelial binding of rhinovirus-C and risk for lung or sinus pathology. In order to further delineate a role for CDHR3 in OM, we performed the following: exome sequencing using DNA samples from OM-affected individuals from 257 multi-ethnic families; Sanger sequencing, logistic regression and transmission disequilibrium tests for 407 US trios or probands with OM; 16S rRNA sequencing and analysis for middle ear and nasopharyngeal samples; and single-cell RNA sequencing and differential expression analyses for mouse middle ear. From exome sequence data, we identified a novel pathogenic CDHR3 splice variant that co-segregates with OM in US and Finnish families. Additionally, a frameshift and six missense rare or low-frequency variants were identified in Finnish probands. In US probands, the CDHR3 p.Cys529Tyr variant was associated with the absence of middle ear fluid at surgery and also with increased relative abundance of Lysobacter in the nasopharynx and Streptomyces in the middle ear. Consistent with published data on airway epithelial cells and our RNA-sequence data from human middle ear tissues, Cdhr3 expression is restricted to ciliated epithelial cells of the middle ear and is downregulated after acute OM. Overall, these findings suggest a critical role for CDHR3 in OM susceptibility. KEY MESSAGES: ⢠Novel rare or low-frequency CDHR3 variants putatively confer risk for otitis media. ⢠Pathogenic variant CDHR3 c.1653 + 3G > A was found in nine families with otitis media. ⢠CDHR3 p.Cys529Tyr was associated with lack of effusion and bacterial otopathogens. ⢠Cdhr3 expression was limited to ciliated epithelial cells in mouse middle ear. ⢠Cdhr3 was downregulated 3 h after infection of mouse middle ear.
Subject(s)
Cadherin Related Proteins/genetics , Membrane Proteins/genetics , Otitis Media/genetics , Animals , Child , Child, Preschool , Female , Genetic Predisposition to Disease , Humans , Infant , Male , Mice, Inbred C57BL , Microbiota/genetics , Mutation , Otitis Media/microbiology , RNA, Ribosomal, 16S , TranscriptomeABSTRACT
OBJECTIVE: To characterize the upper and lower airway findings in children with Down syndrome and chronic respiratory symptoms, based on evaluation by flexible bronchoscopy (FB) with bronchoalveolar lavage and microlaryngoscopy with bronchoscopy (MLB). STUDY DESIGN: A retrospective review was conducted of children with Down syndrome aged 1 month to 17 years, who underwent both FB and MLB within a 1-year timeframe between 2010 and 2019 at Children's Hospital Colorado. Anatomic airway findings are reported as frequencies within the cohort. Bronchoalveolar lavage fluid (BALF) culture results, cell differential, and cytopathology are reported as frequencies or mean ± standard deviation. BALF results were compared between children with and without dysphagia documented on a recent swallow evaluation. RESULTS: Overall, 168 children with Down syndrome were included, with median age of 2.1 years (interquartile range: 0.9-5.1 years). At least one abnormal airway finding was recorded in 96% of patients and 46% had at least three abnormal findings. The most common findings included tracheomalacia (39% FB; 37% MLB), subglottic stenosis (35% MLB), pharyngomalacia (32% FB), and laryngomalacia (16% FB; 30% MLB). Comparison of BALF based on dysphagia status showed that children with dysphagia had more frequent cultures positive for mixed upper respiratory flora (76% vs. 47%, p = 0.004) and a higher percentage of neutrophils (20% vs. 7%, p = 0.006). CONCLUSION: Abnormal findings for FB and MLB are common in children with Down syndrome and chronic respiratory symptoms, and performing the procedures together may increase the diagnostic yield.
Subject(s)
Airway Obstruction , Down Syndrome , Airway Obstruction/diagnosis , Airway Obstruction/etiology , Bronchoscopy , Child , Child, Preschool , Down Syndrome/complications , Humans , Infant , Inflammation/complications , Retrospective StudiesABSTRACT
Otitis media (OM) is a leading cause of childhood hearing loss. Variants in FUT2, which encodes alpha-(1,2)-fucosyltransferase, were identified to increase susceptibility to OM, potentially through shifts in the middle ear (ME) or nasopharyngeal (NP) microbiotas as mediated by transcriptional changes. Greater knowledge of differences in relative abundance of otopathogens in carriers of pathogenic variants can help determine risk for OM in patients. In order to determine the downstream effects of FUT2 variation, we examined gene expression in relation to carriage of a common pathogenic FUT2 c.461G>A (p.Trp154*) variant using RNA-sequence data from saliva samples from 28 patients with OM. Differential gene expression was also examined in bulk mRNA and single-cell RNA-sequence data from wildtype mouse ME mucosa after inoculation with non-typeable Haemophilus influenzae (NTHi). In addition, microbiotas were profiled from ME and NP samples of 65 OM patients using 16S rRNA gene sequencing. In human carriers of the FUT2 variant, FN1, KMT2D, MUC16 and NBPF20 were downregulated while MTAP was upregulated. Post-infectious expression in the mouse ME recapitulated these transcriptional differences, with the exception of Fn1 upregulation after NTHi-inoculation. In the NP, Candidate Division TM7 was associated with wildtype genotype (FDR-adj-p=0.009). Overall, the FUT2 c.461G>A variant was associated with transcriptional changes in processes related to response to infection and with increased load of potential otopathogens in the ME and decreased commensals in the NP. These findings provide increased understanding of how FUT2 variants influence gene transcription and the mucosal microbiota, and thus contribute to the pathology of OM.
Subject(s)
Fucosyltransferases , Haemophilus Infections , Microbiota , Nasopharynx , Otitis Media , Animals , Ear, Middle , Fucosyltransferases/genetics , Haemophilus Infections/metabolism , Haemophilus influenzae/genetics , Humans , Mice , Microbiota/genetics , Nasopharynx/microbiology , Otitis Media/genetics , Otitis Media/metabolism , RNA, Ribosomal, 16S/genetics , Galactoside 2-alpha-L-fucosyltransferaseABSTRACT
BACKGROUND: Esophageal atresia with tracheoesophageal fistula (EA/TEF) is associated with many congenital and vascular malformations; however, reports utilizing computed tomography (CT) and computed tomography angiography (CTA) are limited. The objective of this study is to review CT scans of the chest from patients with EA/TEF and report their pulmonary and vascular findings. METHODS: We completed a retrospective chart review of children with congenital EA/TEF evaluated in the aerodigestive clinic at Children's Hospital Colorado. Results of the most recent CTA or CT of the chest were investigated. Demographics, medical conditions, and bronchoscopy findings were also recorded. The ratio of tracheal lumen area between inspiratory and expiratory CTA images was measured. RESULTS: Of the patients with congenital EA/TEF seen in the program, 47 patients had a chest CT available for review. Eight patients (17%) had bronchiectasis. Of the contrast CT scans, 15 (58%) had a vascular abnormality and 16 (62%) demonstrated tracheal compression (38% at the level of the innominate artery, 35% from other structures). Nineteen of the CTAs had volumetric expiratory images of the trachea to evaluate tracheomalacia. The mean expiratory:inspiratory area was 0.57 (SD ± 0.23) at the level of the innominate. CONCLUSION: Patients with EA/TEF frequently have vascular abnormalities that may alter airway mechanics as well as pulmonary comorbidities that may affect long-term management. For patients experiencing persistent respiratory symptoms, CTA of the chest should be considered adjunct to bronchoscopy to help with medical and surgical management of these children.
Subject(s)
Bronchiectasis/epidemiology , Esophageal Atresia/epidemiology , Tracheoesophageal Fistula/epidemiology , Vascular Diseases/epidemiology , Bronchiectasis/diagnostic imaging , Bronchoscopy , Child , Child, Preschool , Comorbidity , Esophageal Atresia/diagnostic imaging , Female , Humans , Lung/diagnostic imaging , Male , Tomography, X-Ray Computed , Trachea/diagnostic imaging , Tracheoesophageal Fistula/diagnostic imaging , Vascular Diseases/diagnostic imagingABSTRACT
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.
Subject(s)
Otolaryngology , Tracheostomy , Child , Consensus , Humans , Infant , Tracheostomy/adverse effectsABSTRACT
Otitis media (OM), a very common disease in young children, can result in hearing loss. In order to potentially replicate previously reported associations between OM and PLG, exome and Sanger sequencing, RNA-sequencing of saliva and middle ear samples, 16S rRNA sequencing, molecular modeling, and statistical analyses including transmission disequilibrium tests (TDT) were performed in a multi-ethnic cohort of 718 families and simplex cases with OM. We identified four rare PLG variants c.112A > G (p.Lys38Glu), c.782G > A (p.Arg261His), c.1481C > T (p.Ala494Val) and c.2045 T > A (p.Ile682Asn), and one common variant c.1414G > A (p.Asp472Asn). However TDT analyses for these PLG variants did not demonstrate association with OM in 314 families. Additionally PLG expression is very low or absent in normal or diseased middle ear in mouse and human, and salivary expression and microbial α-diversity were non-significant in c.1414G > A (p.Asp472Asn) carriers. Based on molecular modeling, the novel rare variants particularly c.782G > A (p.Arg261His) and c.2045 T > A (p.Ile682Asn) were predicted to affect protein structure. Exploration of other potential disease mechanisms will help elucidate how PLG contributes to OM susceptibility in humans. Our results underline the importance of following up findings from genome-wide association through replication studies, preferably using multi-omic datasets.
Subject(s)
Mutation, Missense , Otitis Media/genetics , Plasminogen/genetics , Animals , Ear, Middle/metabolism , Ear, Middle/microbiology , Female , Genomics/methods , Humans , Male , Mice , Microbiota , Otitis Media/microbiology , Otitis Media/pathology , Pedigree , Plasminogen/metabolism , Polymorphism, Single Nucleotide , Saliva/metabolismABSTRACT
INTRODUCTION: Myringoplasties are common pediatric procedures used to surgically close a perforated tympanic membrane. While a wide variety of graft materials are available to surgeons, the cost effectiveness of these different techniques is not well studied. OBJECTIVES: To compare the cost effectiveness of the fat graft myringoplasty (FGM) with the hyaluronic acid fat graft myringoplasty (HAFGM). METHODS: Retrospective chart review of patients ages 31 days to 18 years who had undergone either FGM or HAFGM from 2006 to 2016. RESULTS: We identified 85 patients who had undergone FGM and 51 patients who had undergone HAFGM. The two groups were statistically similar in age (CI -0.51, 1.9; p = 0.23), sex (CI 0.3, 1.4; p = 0.27), and history of prior tympanostomy tube placement (CI -0.07, 0.07; p = 0.69). Both groups had a similar number of total comorbidities (40.0% of patients in the FGM group and 27.5% of patients in the HAFGM; CI -0.04, 0.29; p = 0.19). The FGM and the HAFGM procedure did not have statistically significant differences in perforation closure rates, 82.4% and 92.2% respectively (CI 0.81, 7.3; p = 0.13). In comparing the total surgeons' cost of closing a tympanic membrane deficit, the FGM incurred a greater cost per perforation. The cost/tympanic membrane perforation closure for the FGM totaled $3011.88 per deficit, whereas the HAFGM totaled $2742.98. CONCLUSION: As financial stewardship becomes more important for medical decision making, it is imperative that providers consider cost and outcomes data together when comparing similar treatment options. The FGM and the HAFGM have statistically similar rates of success in closing tympanic membrane perforations. In this study, the FGM cohort consumed more health care dollars per perforation secondary to the need for revision surgeries. As such, this study offers that the additional use of a hyaluronic disc does not increase overall cost to the healthcare system when performing a fat graft myringoplasty over a large cohort of patients.
Subject(s)
Adipose Tissue/transplantation , Direct Service Costs , Hyaluronic Acid/therapeutic use , Myringoplasty/methods , Tympanic Membrane/surgery , Adolescent , Child , Child, Preschool , Female , Humans , Hyaluronic Acid/economics , Infant , Male , Myringoplasty/economics , Retrospective Studies , Treatment Outcome , Tympanic Membrane Perforation/surgeryABSTRACT
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.
Subject(s)
Betacoronavirus , Bronchoscopy , Coronavirus Infections/prevention & control , Laryngoscopy , Pandemics/prevention & control , Pneumonia, Viral/prevention & control , COVID-19 , Child , Clinical Protocols , Humans , Perioperative Period , SARS-CoV-2ABSTRACT
OBJECTIVE: The Clinical Assessment Score-15 (CAS-15) has been validated as an office-based assessment for pediatric sleep-disordered breathing in otherwise healthy children. Our objective was to determine the generalizability of the CAS-15 in a multi-institutional fashion. METHODS: Five hundred and thirty children from 13 sites with suspected sleep-disordered breathing were recruited, and the investigators completed the CAS-15. Based on decisions made in the course of clinical care, investigators recommended overnight polysomnography, observation, medical therapy, and/or surgery. Two hundred and forty-seven subjects had a follow-up CAS-15. RESULTS: Mean age was 5.1 (2.6) years; 54.2% were male; 39.1% were white; and 37.0% were African American. Initial mean (standard deviation [SD]) CAS-15 was 37.3 (12.7), n = 508. Spearman correlation between the initial CAS-15 and the initial apnea-hypopnea index (AHI) was 0.41 (95% confidence interval [CI], 0.29, 0.51), n = 212, P < .001. A receiver-operating characteristic curve predicting positive polysomnography (AHI > 2) had an area under the curve of 0.71 (95% CI, 0.63, 0.80). A score ≥ 32 had a sensitivity of 69.0% (95% CI, 61.7, 75.5), a specificity of 63.4% (95% CI, 47.9, 76.6), a positive predictive value of 88.7% (95% CI, 82.1, 93.1), and a negative predictive value of 32.9% (95% CI, 23.5, 44.0) in predicting positive polysomnography. Among children who underwent surgery, the mean change (SD) score was 30.5 (12.6), n = 201, t = 36.85, P < .001, effect size = 3.1. CONCLUSION: This study establishes the generalizability of the CAS-15 as a useful office tool for the evaluation of pediatric sleep-disordered breathing. LEVEL OF EVIDENCE: 2B Laryngoscope, 130:2256-2262, 2020.
Subject(s)
Polysomnography/statistics & numerical data , Severity of Illness Index , Sleep Apnea Syndromes/diagnosis , Symptom Assessment/statistics & numerical data , Child , Child, Preschool , Female , Humans , Male , Polysomnography/methods , Predictive Value of Tests , ROC Curve , Reproducibility of Results , Sensitivity and Specificity , Statistics, Nonparametric , Symptom Assessment/methodsABSTRACT
OBJECTIVES/HYPOTHESIS: The Aerodigestive Program (the Aero Program) at Children's Hospital Colorado is a multidisciplinary program focused on airway, digestive, and lung disorders in complex children, involving collaboration between gastroenterology, pulmonology, anesthesiology, and otolaryngology in clinic and operating room. These programs have proliferated as institutions focus on providing greater care coordination and family satisfaction. However, few cost, charge, and satisfaction data exist to support these resource-intensive programs. The goal of this study was to investigate the value of combined triple endoscopy delivered by the Aero Program through analysis of institutional charges, direct costs, operating room efficiency metrics, and parent satisfaction. STUDY DESIGN: Program evaluation. METHODS: Finance, satisfaction, efficiency, and quality-of-care metrics were evaluated within and outside of the Aero Program through retrospective queries of electronic health records, administrative databases, and parent surveys at our institution. RESULTS: Mean anesthesia time in the Aero Program was 54 minutes (49-60; 95% confidence interval), which was significantly less (P < .0001) than the estimated 89 minutes of having the three procedures done separately. Average charges and average direct costs for triple endoscopy were 38.8% and 41.9% less than the sum of the averages for separate procedures, respectively. Parent satisfaction was high for the Aero Program care. CONCLUSIONS: As organizations move toward greater coordination of care for complex patients, multidisciplinary programs must demonstrate their value by delivering cost-effective care. Aerodigestive programs have the potential to provide satisfying care that is less costly to the organization, insurer, and family. These programs represent a step in the evolution toward higher value care and value-based payment methodology. LEVEL OF EVIDENCE: 4 Laryngoscope, 130:521-525, 2020.
Subject(s)
Delivery of Health Care, Integrated/organization & administration , Digestive System Diseases/therapy , Otorhinolaryngologic Diseases/therapy , Respiratory Tract Diseases/therapy , Colorado , Efficiency, Organizational , Gastroenterology , Hospitals, Pediatric , Humans , Otolaryngology , Patient Satisfaction , Program Evaluation , Pulmonary Medicine , Quality of Health Care , Retrospective Studies , Speech-Language PathologyABSTRACT
OBJECTIVE: To determine the frequency and risk factors that lead to the development of persistent TCF (tracheocutaneous fistula) formation in children following tracheostomy decannulation at our institution. METHODS: A retrospective chart review of all pediatric patients at Children's Hospital Colorado who underwent tracheostomy decannulation and were being followed between January 1, 2007 and December 31, 2013. TCF was defined as a persistent fistula six months following decannulation. We determined patient demographics, age at tracheotomy, primary indication for tracheotomy, tracheostomy-tube size, medical comorbidities, age at decannulation, date of TCF closure, and method of TCF closure. RESULTS: One hundred twenty-nine patients ranging from 51 days to 19 years of age underwent tracheostomy decannulation. 63 (49%) patients underwent surgical closure of TCF. Compared to those with spontaneous closure by multivariable analysis, those with surgical closure were younger at tracheostomy placement (pâ¯=â¯0.0002), had a tracheostomy for a longer duration (pâ¯=â¯0.0025), and were diagnosed with tracheobronchomalacia (pâ¯=â¯0.0051). The likelihood of spontaneous closure decreased over time. Tracheostomy tube internal diameter correlated with age (Râ¯=â¯0.64, pâ¯<â¯0.0001). CONCLUSIONS: Approximately 50% of pediatric tracheostomy stoma sites will close spontaneously. Development of a persistent TCF was associated with younger age at placement, longer duration of tracheostomy, and the presence of tracheobronchomalacia. These observations may help clinicians anticipate outcomes following tracheostomy decannulation in children.
Subject(s)
Airway Extubation/adverse effects , Cutaneous Fistula/surgery , Postoperative Complications/surgery , Respiratory Tract Fistula/surgery , Tracheal Diseases/surgery , Tracheostomy/adverse effects , Adolescent , Child , Child, Preschool , Colorado , Cutaneous Fistula/etiology , Device Removal/adverse effects , Female , Hospitals, Pediatric , Humans , Infant , Male , Postoperative Complications/etiology , Respiratory Tract Fistula/etiology , Retrospective Studies , Risk Factors , Time Factors , Tracheal Diseases/etiology , Tracheobronchomalacia/surgery , Tracheotomy/adverse effects , Young AdultABSTRACT
Background: Posterior tracheomalacia is characterized by collapsibility of the posterior trachea and is often present in patients with congenital esophageal atresia (EA) with or without tracheoesophageal fistula (TEF). It can lead to a variety of symptoms from mild expiratory stridor and difficulty clearing secretions to severe respiratory distress, especially in the setting of increased work of breathing. Depending on the severity of symptoms, treatment ranges from medical therapy, including airway clearance techniques, aerosolized medications, and steroids to surgical treatment. The purpose of this study is to review our institution's experience with posterior tracheopexy. Materials and Methods: A retrospective review was conducted from 2017-2019 at a freestanding quaternary care children's hospital. Results: The analysis included 8 patients. The median age at surgery was 6 (range 3-8) years and 4 (50%) of cohort were male. The majority of patients (n = 6, 75%) had a history of prior EA and TEF repair and 3 (38%) had associated VACTERL anomalies. All patients demonstrated severe tracheomalacia on preoperative bronchoscopy with collapse of the posterior membrane. In regard to surgical approach, most cases (6/8, 75%) underwent thoracoscopic repair. The median operative time was 218 (193 thoracoscopic, 218 open) minutes. The median length of stay was 3 days, and 2 (25%) patients had a postoperative complication of chylothorax (1 thoracoscopic and 1 open), both of which resolved without invasive intervention. The current median length of follow-up is 3 months, and all patients reported symptomatic improvement. One patient who had initial symptomatic improvement has undergone repeat tracheopexy for recurrence. Conclusion: Posterior tracheopexy is an effective treatment option for symptoms associated with tracheomalacia. The thoracoscopic approach is feasible in experienced hands and with the support of a multidisciplinary team.
Subject(s)
Thoracoscopy/methods , Trachea/surgery , Tracheomalacia/surgery , Bronchoscopy , Child , Child, Preschool , Feasibility Studies , Female , Follow-Up Studies , Humans , Male , Recurrence , Retrospective Studies , Trachea/diagnostic imaging , Tracheomalacia/diagnostic imaging , Treatment OutcomeABSTRACT
A genetic basis for otitis media is established, however, the role of rare variants in disease etiology is largely unknown. Previously a duplication variant within A2ML1 was identified as a significant risk factor for otitis media in an indigenous Filipino population and in US children. In this report exome and Sanger sequencing was performed using DNA samples from the indigenous Filipino population, Filipino cochlear implantees, US probands, Finnish, and Pakistani families with otitis media. Sixteen novel, damaging A2ML1 variants identified in otitis media patients were rare or low-frequency in population-matched controls. In the indigenous population, both gingivitis and A2ML1 variants including the known duplication variant and the novel splice variant c.4061 + 1 G>C were independently associated with otitis media. Sequencing of salivary RNA samples from indigenous Filipinos demonstrated lower A2ML1 expression according to the carriage of A2ML1 variants. Sequencing of additional salivary RNA samples from US patients with otitis media revealed differentially expressed genes that are highly correlated with A2ML1 expression levels. In particular, RND3 is upregulated in both A2ML1 variant carriers and high-A2ML1 expressors. These findings support a role for A2ML1 in keratinocyte differentiation within the middle ear as part of otitis media pathology and the potential application of ROCK inhibition in otitis media.