Alternatives to Polysomnography for the Diagnosis of Pediatric Obstructive Sleep Apnea.

Diagnosis of obstructive sleep apnea (OSA) in children with sleep-disordered breathing (SDB) requires hospital-based, overnight level I polysomnography (PSG). Obtaining a level I PSG can be challenging for children and their caregivers due to the costs, barriers to access, and associated discomfort. Less burdensome methods that approximate pediatric PSG data are needed. The goal of this review is to evaluate and discuss alternatives for evaluating pediatric SDB. To date, wearable devices, single-channel recordings, and home-based PSG have not been validated as suitable replacements for PSG. However, they may play a role in risk stratification or as screening tools for pediatric OSA. Further studies are needed to determine if the combined use of these metrics could predict OSA.

Long-Term Outcomes of Tracheostomy-Dependent Children.

OBJECTIVE: To estimate the 1-, 5-, and 10-year survival and decannulation rates of children with a tracheostomy. STUDY DESIGN: Ambidirectional cohort. SETTING: Tertiary children's hospital. METHODS: All patients (<18 years) that had a tracheostomy placed between 2009 and 2020 were included and followed until 21 years of age, decannulation, or death. The Kaplan-Meier method estimated cumulative probabilities of death and decannulation. RESULTS: A total of 551 children underwent tracheostomy at a median age of 7.2 months (interquartile range [IQR]: 3.8-49.2). Children were followed for a median of 2.1 years (IQR: 0.7-4.2, range 0-11.5). The cumulative probability of mortality at 1 year was 11.9% (95% confidence interval [CI]: 9.4-15.1), at 5 years was 26.1% (95% CI: 21.6-31.3), and at 10 years was 41.6% (95% CI: 32.7-51.8). Ventilator dependence at index discharge (hazard ratio [HR]: 2.04, 95% CI: 1.10-3.81, p = .03), severe neurologic disability (HR: 2.79, 95% CI: 1.61-4.84, p < .001), and cardiac disease (HR: 1.69, 95% CI: 1.08-2.65, p = .02) were associated with time to death. The cumulative probability of decannulation was 10.4% (95% CI: 8.0-13.5), 44.9% (95% CI: 39.4-50.9), and 54.1% (95% CI: 47.4-61.1) at 1 year, 5 years, and 10 years, respectively. Ventilator dependence (HR: 0.43, 95% CI: 0.31-0.60, p < .001), severe neurologic disability (HR: 0.20, 95% CI: 0.14-0.30, p < .001), and tracheostomy indicated for respiratory failure (HR: 0.68, 95% CI: 0.48-0.96, p = .03) correlated with longer decannulation times. CONCLUSION: After tracheostomy, estimated mortality approaches 42% by 10 years and decannulation approaches 54%. Children with ventilator support at discharge and severe neurological disability had poorer long-term survival and longer times to decannulation.

Resting-State Cerebral Hemodynamics is Associated With Problem Behaviors in Pediatric Sleep-Disordered Breathing.

OBJECTIVE: Untreated sleep-disordered breathing (SDB) is associated with problem behaviors in children. The neurological basis for this relationship is unknown. We used functional near-infrared spectroscopy (fNIRS) to assess the relationship between cerebral hemodynamics of the frontal lobe of the brain and problem behaviors in children with SDB. STUDY DESIGN: Cross-sectional. SETTING: Urban tertiary care academic children's hospital and affiliated sleep center. METHODS: We enrolled children with SDB aged 5 to 16 years old referred for polysomnography. We measured fNIRS-derived cerebral hemodynamics within the frontal lobe during polysomnography. We assessed parent-reported problem behaviors using the Behavioral Response Inventory of Executive Function Second Edition (BRIEF-2). We compared the relationships between (i) the instability in cerebral perfusion in the frontal lobe measured fNIRS, (ii) SDB severity using apnea-hypopnea index (AHI), and (iii) BRIEF-2 clinical scales using Pearson correlation (r). A p < .05 was considered significant. RESULTS: A total of 54 children were included. The average age was 7.8 (95% confidence interval, 7.0-8.7) years; 26 (48%) were boys and 25 (46%) were Black. The mean AHI was 9.9 (5.7-14.1). There is a statistically significant inverse relationship between the coefficient of variation of perfusion in the frontal lobe and BRIEF-2 clinical scales (range of r = 0.24-0.49, range of p = .076 to <.001). The correlations between AHI and BRIEF-2 scales were not statistically significant. CONCLUSION: These results provide preliminary evidence for fNIRS as a child-friendly biomarker for the assessment of adverse outcomes of SDB.

Tracheostomy in the extremely premature neonate - Long term outcomes in a multi-institutional study.

OBJECTIVES: To describe the long-term outcomes related to breathing, feeding, and neurocognitive development in extremely premature infants requiring tracheostomy. STUDY DESIGN: Pooled cross-sectional survey. SETTING: Multi-institutional academic children's hospitals. METHODS: Extremely premature infants who underwent tracheostomy between January 1, 2012, and December 31, 2019, at four academic hospitals were identified from an existing database. Information was gathered from responses to a questionnaire by caregivers regarding airway status, feeding, and neurodevelopment 2-9 years after tracheostomy. RESULTS: Data was available for 89/91 children (96.8%). The mean gestational age was 25.5 weeks (95% CI 25.2-25.7) and mean birth weight was 0.71 kg (95% CI 0.67-0.75). Mean post gestational age at tracheostomy was 22.8 weeks (95% CI 19.0-26.6). At time of the survey, 18 (20.2%) were deceased. 29 (40.8%) maintained a tracheostomy, 18 (25.4%) were on ventilatory support, and 5 (7%) required 24-h supplemental oxygen. Forty-six (64.8%) maintained a gastrostomy tube, 25 (35.2%) had oral dysphagia, and 24 (33.8%) required a modified diet. 51 (71.8%) had developmental delay, 45 (63.4%) were enrolled in school of whom 33 (73.3%) required special education services. CONCLUSIONS: Tracheostomy in extremely premature neonates is associated with long term morbidity in the pulmonary, feeding, and neurocognitive domains. At time of the survey, about half are decannulated, with a majority weaned off ventilatory support indicating improvement in lung function with age. Feeding dysfunction is persistent, and a significant number will have some degree of neurocognitive dysfunction at school age. This information may help caregivers regarding expectations and plans for resource management.

Pathogenic bacteria in bronchoalveolar lavage cultures and pediatric laryngotracheal reconstruction outcomes.

OBJECTIVE: The primary objective was to determine if treating pathogenic bacteria in bronchoalveolar lavage (BAL) cultures improves outcomes after pediatric double stage laryngotracheal reconstruction (dsLTR). STUDY DESIGN: Case series with chart review. SETTING: Tertiary children's hospital. METHODS: All children (<18 years) obtaining flexible bronchoscopy with BAL cultures before dsLTR between 2016 and 2022 were included. Cultures identified abnormal bacterial growth or normal respiratory flora. Thirty-day postoperative surgical site or lung infections were captured and tracheostomy decannulation rates were obtained for children with at least 12 months of follow-up. RESULTS: Twenty-seven children obtained presurgical BAL cultures before dsLTR. Median age at reconstruction was 2.9 years (interquartile range: 2.3-3.5) and 89% (24/27) had high grade subglottic stenosis. Positive cultures were obtained in 56% of children (N = 15) with Pseudomonas aeruginosa (40%, 6/15) and methicillin-resistant Staphylococcus aureus (33%, 5/15) the most frequent organisms. All children with positive cultures were treated based on culture and sensitivity data. Postoperative infections developed in 22% (6/27) of children with equal distribution of surgical site and respiratory infections among children with pathogenic bacteria and normal respiratory flora. At 12 months after surgery, the decannulation rate was no different between those treated and not treated for a presurgical positive BAL culture (47% vs. 58%, p = 0.70). CONCLUSION: Pathogenic bacteria are common in BAL cultures from tracheostomy-dependent children before dsLTR. Treatment keeps respiratory infections and decannulation rates similar to children with negative cultures, suggesting continued benefit of flexible bronchoscopy and BAL in preparation for these surgeries.

Incidence of Persistent Tracheocutaneous Fistula After Pediatric Tracheostomy Decannulation.

OBJECTIVES: To determine the incidence of tracheocutaneous fistula (TCF) and identify characteristics associated with persistence. STUDY DESIGN: Prospective cohort. METHODS: All successfully decannulated children (<18 years) between 2014 and 2020 at a tertiary children's hospital were included. Revision tracheostomies, concomitant major neck surgery, or single-stage laryngotracheal reconstructions were excluded. A persistent TCF was defined as a patent fistula at 6 weeks after decannulation. RESULTS: A total of 77 children met inclusion criteria with a persistent TCF incidence of 65% (50/77). Children with a persistent TCF were younger at placement (1.4 years (SD: 3.3) vs. 8.5 years (SD: 6.5), p < 0.001) and tracheostomy-dependent longer (2.8 years (SD: 1.3) vs. 0.9 years (SD: 0.7), p < 0.001). On univariate analysis, placement under 12 months of age (86% vs. 26% p < 0.001), duration of tracheostomy more than 2 years (76% vs. 11% p < 0.001), short gestation (64% vs. 26%, p = 0.002), congenital malformations (64% vs. 33%, p = 0.02), newborn complications (58% vs. 26%, p = 0.009), maternal complications (40% vs. 11%, p = 0.009) and chronic respiratory failure (72% vs. 41%, p = 0.01) were associated with persistent TCF. Logistic regression analysis associated duration of tracheostomy (OR: 0.14, 95% CI: 0.05-0.35, p < 0.001) and congenital malformations (OR: 0.25, 95% CI: 0.06-0.99, p = 0.049) with failure to spontaneously close. CONCLUSIONS: Two-thirds of children will develop a persistent TCF after tracheostomy decannulation. Persistent TCF is correlated with a longer duration of tracheostomy and congenital malformations. Anticipation of this event in higher-risk children is necessary when caring for pediatric tracheostomy patients. LEVEL OF EVIDENCE: 3 Laryngoscope, 133:417-422, 2023.

Mortality Among Children with a Tracheostomy.

OBJECTIVES: To characterize the cause of death among children with a tracheostomy. STUDY DESIGN: Prospective cohort. METHODS: All pediatric patients (<18 years) who had a tracheostomy placed at a tertiary care institution between 2015 and 2020 were included. The location and cause of death were recorded along with patient demographics and age. RESULTS: A total of 271 tracheostomies were placed with 46 mortalities reviewed for a mortality rate of 16.8%. Mean age at placement was 1.7 years (SD: 3.4) and mean age at death was 2.9 years (SD: 3.5). Most tracheostomies were placed for respiratory failure (N = 33, 72%). The mean time to death after tracheostomy was 1.2 years (SD: 1.2) and 28% (N = 13) occurred during the same admission as placement. Mean time to death after hospital discharge was 1.3 years (SD: 1.3). Etiology of death was respiratory failure (33%, N = 15), cardiopulmonary arrest (15%, N = 7), unknown (43%, N = 20), or secondary to a tracheostomy-related complication for 9% (N = 4). Location of death was in intensive care units for 41% (N = 19) and 30% died at home (N = 14). Comfort care measures were taken for 37% (N = 17). Severe neurological disability (HR: 4.06, p = 0.003, 95% CI: 1.59-10.34) and congenital heart disease (HR: 2.36, p = 0.009, 95% CI: 1.24-4.48) correlated with time to death on Cox proportional hazard modeling. CONCLUSIONS: Nearly one-third of children with a tracheostomy who expire will do so during the same admission as tracheostomy placement. Although progression of underlying disease will lead to most deaths, 9% will be a result of a tracheostomy-related complication, which represents a meaningful target for quality improvement initiatives. LEVEL OF EVIDENCE: 3 Laryngoscope, 133:403-409, 2023.

Emergency Department Visits and Hospitalizations After Pediatric Tracheostomy.

OBJECTIVE: To determine factors associated with frequent emergency department (ED) visits and hospitalizations after pediatric tracheostomy. METHODS: A prospective cohort of children (<18 years) with a tracheostomy placed at a tertiary children's hospital between 2015 and 2019 were followed for 24 months after index discharge. ED visits and hospitalizations were recorded to identify risk factors for frequent utilization (≥4 visits). RESULTS: A total of 239 children required 1285 total visits to the ED or hospital after index discharge with 112 children (47%) having ≥4 visits. Respiratory-related illness was the most common reason (N = 699, 54%) followed by gastrostomy tube issues (N = 119, 9.3%). Frequent utilization was associated with Black race (OR: 2.01, 95% CI: 1.18-3.70, p = 0.01), mechanical ventilation (OR: 2.74, 95% CI: 1.35-5.59, p = 0.006), and Spanish language (OR: 3.86, 95% CI: 1.47-10.11, p = 0.006) on regression modeling. There were no predictors of visits for tracheostomy-related complications, which accounted for 4.8% of all encounters. A sub-analysis showed that Hispanic ethnicity and gestational age predicted visits for respiratory failure. CONCLUSION: Frequent ED visits or hospitalizations are required for 47% of children in the first 2 years after tracheostomy placement. Ventilatory support, Black race, and Spanish language increase the likelihood of high utilization. Although tracheostomy-related visits are uncommon, strategies to anticipate and decrease respiratory-related admissions may have the most impact. LEVEL OF EVIDENCE: 3 Laryngoscope, 133:2018-2024, 2023.

Tracheostomy on infants born in the periviable period: Outcomes at discharge from the neonatal intensive care unit (NICU).

OBJECTIVES: Advances in neonatal intensive care have allowed successful resuscitation of children born at the border of viability. However, there has been little change in the incidence of bronchopulmonary dysplasia (BPD) and anatomical upper airway obstruction which may require a tracheostomy in that group. The benefits of the procedure are accompanied by sequelae that impact outcomes. Information about these issues can assist caregivers in making decisions and planning care after discharge from the neonatal intensive care unit (NICU). The objectives of this study were to describe the clinical characteristics of neonates born in the periviable period (≤25 weeks gestation) requiring tracheotomy and to highlight their hospital course, complications and status upon NICU discharge. METHODS: Retrospective analysis at four tertiary care academic children's hospitals. Medical records of neonates born ≤25 weeks gestation who required tracheotomy between January 1, 2012 and December 31, 2018 were reviewed. Demographics, medical comorbidities, and tracheostomy related complications were studied. Feeding, ventilation, and neurodevelopmental outcomes at time of transfer from NICU were evaluated. RESULTS: Fifty-two patients were included. The mean gestational age was 24.3 (95% confidence interval, 24.1 to 24.5) weeks. The mean birth weight was 635 (95% CI: 603 to 667) grams and 50 (96.2%) children had BPD. At time of discharge from the NICU, 47 (90.4%) required mechanical ventilation, four (7.7%) required supplemental oxygen and one (1.9%) was weaned to room air. Forty-two (80.8%) were discharged with a gastrostomy tube, seven (28%) with a nasogastric tube, and three (5.8%) were on oral feeds. Two (3.8%) suffered hypoxic ischemic encephalopathy, 27 (51.9%) had neurodevelopmental delay, seven (13.5%) were diagnosed with another anomaly, and 16 (30.8%) were considered normal. Complications related to the procedure were observed in 28 (53.8%) neonates. Granulation tissue was seen in 17 (32.7%), wound break down or cellulitis in three (5.8%), one (1.9%) with tracheostomy plugging, three (5.8%) with dislodgement of the tracheostomy tube and four (7.7%) developed tracheitis. CONCLUSIONS: Tracheostomy in infants born in the periviable period is primarily performed for BPD and portends extended ventilatory dependence. It is associated with non-oral alimentation at the time of discharge from the NICU and developmental delay. Mortality directly related to the procedure is rare. Minor complications are common but do not require surgical intervention. These data may aid in counseling caregivers about the procedure in this vulnerable population.

Adenochondroma of the Thyroid in a 3-Year-Old Female: A Case Report.

Follicular adenomas are the most common benign thyroid neoplasm but are unusual in children. However, other rare degenerative lesions and those of developmental origin can also present as thyroid masses. This article reports the first described pediatric thyroid adenochondroma. A 3-year-old female presented with a hard mass in the right lobe of her thyroid with nondiagnostic imaging and cytology findings. She underwent a right thyroid lobectomy uneventfully. Final histopathology examination confirmed an adenochondroma. To the best of our knowledge, an adenochondroma of the thyroid gland in a child has not been previously reported in literature. Though a rare and benign entity, thyroid adenochondromas present clinically with many features concerning for malignancy. Therefore, these lesions should be considered in the differential diagnosis of pediatric thyroid masses.

Predictors of Tracheostomy Decannulation in Adult Laryngotracheal Stenosis.

OBJECTIVE: Predictors of tracheostomy decannulation in patients with laryngotracheal stenosis are not fully known, making prognosis difficult. The aim was to identify predictors of tracheostomy decannulation in adult patients with acquired stenosis of the larynx and/or trachea who were tracheostomy dependent. STUDY DESIGN: Case series. SETTING: Academic teaching hospital. METHODS: A total of 103 consecutive adult patients with laryngotracheal stenosis who were tracheostomy dependent and seen by the otolaryngology clinic from January 1, 2013, to August 2, 2018, were included. Exclusion criteria included age <18 years, history of laryngeal cancer or head and neck radiation, or history of laryngeal fracture. The primary outcome was the presence of tracheostomy at last follow-up. The patients' etiology of stenosis, comorbid conditions, and characteristics of the stenosis were analyzed to determine if there was a statistically significant relationship with decannulation. RESULTS: A total of 103 patients were included: 67% of patients were women and the average age was 53.5 years. Sixty-four patients (62%) were successfully decannulated. In multivariate analysis, patients who were successfully decannulated presented to the otolaryngology clinic earlier after tracheostomy was performed, were more likely to have been intubated due to trauma, and were less likely to have gastroesophageal reflux disease. In patients with subglottic or tracheal stenosis, those with granulation tissue without firm scar were more likely to be decannulated, and those who underwent rigid dilation were less likely to be decannulated. CONCLUSION: Early evaluation by an otolaryngologist may increase the likelihood of tracheostomy decannulation in patients with laryngotracheal stenosis. Patient comorbidities may assist in predicting which patients will be successfully decannulated.

Echocardiographic screening in children with very severe obstructive sleep apnea.

OBJECTIVE: (i) To determine the prevalence of echocardiographic abnormalities in children with very severe OSA defined by an apnea hypopnea index (AHI) ≥ 30 events/hour. (ii) To test the hypothesis that polysomnographic parameters predict echocardiographic variables in this population. METHODS: Children aged 1-17 years presenting with polysomnography demonstrating an AHI ≥30 and referred for pre-operative echocardiography performed within the 6 months prior to tonsillectomy and adenoidectomy (T&A), over a two-year period (January 1, 2016 to December 31, 2018) were evaluated. The exclusion criteria were the presence of (i) unrepaired congenital cardiac disease, (ii) tracheostomy, (iii) poorly controlled asthma, or (iv) neuromuscular disorder. The prevalence of echocardiographic abnormalities was determined for the study population. The impact of the severity of OSA on echocardiographic parameters was evaluated using Student's t-test. The relationships between polysomnographic variables and biventricular function as well as pulmonary hemodynamics were measured. A penalized regression model was used to identify the contributions of polysomnographic variables to each echocardiographic parameter by mitigating inter-variable relationships. P < .05 was considered significant. RESULTS: Eighty-nine children were screened, of whom 47 were included for analysis. The mean age was 68.8 months [95% confidence interval, 56.0 to 81.6]. Thirty-three (70.2%) were boys. Twenty (42.6%) were obese. All children had normal echocardiograms. The differences in echocardiographic variables between children grouped by the severity of OSA were not statistically significant (P: 0.18-0.98). Polysomnographic variables predicted only 4 out of 13 studied echocardiographic parameters. CONCLUSIONS: Pre-operative echocardiography did not identify significant abnormalities in children with very severe OSA. Majority of the echocardiographic variables were not predicted by polysomnographic parameters. This study demonstrates the limited benefit associated with routine echocardiographic screening of children with very severe OSA solely based on polysomnographic indices.

The Role of Cognitive Evaluation in Predicting Successful Audiometric Testing among Children.

OBJECTIVE: To determine the role of cognitive testing in predicting age-appropriate audiometric responses among children aged 30 to 42 months. STUDY DESIGN: Prospective. SETTING: Tertiary care audiology clinic. SUBJECTS AND METHODS: Subjects included primary English-speaking children aged 30 to 42 months. A certified pediatric audiologist performed the cognitive aspect of the Developmental Assessment of Young Children-Second Edition (DAYC-2). A second, blinded audiologist performed age-appropriate audiometry. The raw, age-equivalent, percentile, and standard DAYC-2 scores were compared by agreement between speech reception threshold (SRT) and pure tone average (PTA). Optimal DAYC-2 thresholds were also calculated for prediction of SRT-PTA agreement and assessed for sensitivity, specificity, and positive and negative predictive values. P < .05 was considered significant. RESULTS: Complete data were obtained from 37 children. The mean age was 34.9 months (95% CI, 33.5-36.2), and 15 (41%) were female. Among the 37 children, 24 (65%) and 13 (35%) underwent visual reinforcement audiometry and conditioned play audiometry, respectively. SRT-PTA agreement was seen in 32 (87%) tests. Mean DAYC-2 raw score grouped by SRT-PTA agreement was 39.4 versus 33.4 for nonagreement (2.8-9.3, P < .001). The mean age-equivalent score grouped by SRT-PTA agreement was 29.6 versus 23.0 for nonagreement (2.7-10.6, P = .002). Optimal cut points based on DAYC-2 scores achieved moderate overall prediction performance (area under the curve, 0.73-0.77) with a positive predictive value of 100%. CONCLUSION: The DAYC-2 is a useful screen to identify children likely to complete an age-appropriate audiogram.

Clinical Evaluation of Tinnitus.

The clinical evaluation of patients with tinnitus differs based on whether the tinnitus is subjective or objective. Subjective tinnitus is usually associated with a hearing loss, and therefore, the clinical evaluation is focused on an otologic and audiologic evaluation with adjunct imaging/tests as necessary. Objective tinnitus is divided into perception of an abnormal somatosound or abnormal perception of a normal somatosound. The distinction between these categories is usually possible based on a history, physical examination, and audiogram, leading to directed imaging to identify the underlying abnormality.

Surgical Treatment of Tinnitus.

Surgery for tinnitus can be divided into procedures directed specifically at elimination of tinnitus versus those directed at an independent primary otopathology whose symptoms include tinnitus. For the latter, although there may be an independent primary goal for which the surgery is undertaken, tinnitus may be expected to improve secondarily. This article will address both tinnitus-specific and tinnitus non-specific procedures for objective and subjective causes.

B cells in T follicular helper cell development and function: separable roles in delivery of ICOS ligand and antigen.

B cells are required for follicular Th (Tfh) cell development, as is the ICOS ligand (ICOS-L); however, the separable contributions of Ag and ICOS-L delivery by cognate B cells to Tfh cell development and function are unknown. We find that Tfh cell and germinal center differentiation are dependent on cognate B cell display of ICOS-L, but only when Ag presentation by the latter is limiting, with the requirement for B cell expression of ICOS-L overcome by robust Ag delivery. These findings demonstrate that Ag-specific B cells provide different, yet compensatory, signals for Tfh cell differentiation, while reconciling conflicting data indicating a requirement for ICOS-L expression on cognate B cells for Tfh cell development with those demonstrating that the latter requirement could be bypassed in lieu of that tendered by noncognate B cells. Our findings clarify the separable roles of delivery of Ag and ICOS-L by cognate B cells for Tfh cell maturation and function, and have implications for using therapeutic ICOS blockade in settings of abundantly available Ag, such as in systemic autoimmunity.