Effectiveness of pediatric drug-induced sleep endoscopy for REM-predominant obstructive sleep apnea.

STUDY OBJECTIVES: Because dexmedetomidine (DEX)-induced sedation mimics non-rapid eye movement (NREM) sleep, its utility in sedating children with REM-predominant disease is unclear. We sought to determine the effectiveness of pediatric drug-induced sleep endoscopy (DISE) using DEX and ketamine for children with REM-predominant OSA, specifically whether or not at least one site of obstruction could be identified. METHODS: A retrospective case series of children without tonsillar hypertrophy undergoing DISE at a tertiary pediatric hospital from 10/2013 through 9/2015 who underwent subsequent surgery to address OSA with polysomnography (PSG) before and after. RESULTS: We included 56 children, mean age 5.6±5.4 years, age range 0.1-17.4 years, mean BMI 20.3±7.4 kg/m2 (76±29 percentile). At least one site of obstruction was identified in all patients, regardless of REM- or NREM-predominance. The mean obstructive apnea-hypopnea index (oAHI) improved (12.6 ± 10.7 to 9.0 ± 14.0 events/h) in children with REM-predominant (P = 0.013) and NREM-predominant disease (21.3 ± 18.9 to 10.3 ± 16.2 events/h) (P = 0.008). The proportion of children with a postoperative oAHI < 5 was 53% and 55% for REM- and NREMpredominant OSA, respectively. Unlike children with NREM-predominant disease, children with REM-predominant disease had significant improvement in the mean saturation nadir (P < 0.001), total sleep time (P = 0.006), and sleep efficiency (P = 0.015). CONCLUSIONS: For children with OSA without tonsillar hypertrophy, DISE using DEX/ketamine was useful to predict at least one site of obstruction, even for those with REM-predominant OSA. DISE-directed outcomes resulted in significant improvements in mean oAHI, total sleep time, sleep efficiency, saturation nadir, and the proportion with oAHI < 5, after surgery for some children with REM-predominant disease.

Management of the Disrupted Airway in Children.

OBJECTIVE: Our objective was to gather data that would enable us to suggest more specific guidelines for the management of children with airway disruption. STUDY DESIGN: Retrospective case series with data from five tertiary medical centers. METHODS: Children younger than 18 years of age with a disrupted airway were enrolled in this series. Data pertaining to age, sex, etiology and location of the disruption, type of injury, previous surgery, presence of air extravasation, management, and outcome were obtained and summarized. RESULTS: Twenty children with a mean age of 4.4 years (range 1 day-14.75 years) were included in the study. All were evaluated by flexible endoscopy and/or microlaryngoscopy in the operating room. Twelve (60%) children had tracheal involvement; seven had bronchial involvement; and one had involvement of the cricoid cartilage. Nine children had air extravasation, and all these children required surgical repair. Of the 11 who did not have air extravasation, only one underwent surgical repair. Complete healing of the disrupted airway was seen in all cases. CONCLUSION: This series suggests that if there is no continuous air extravasation demonstrated on imaging studies or clinical examination, nonoperative management may allow for spontaneous healing without sequelae. However, surgical repair may be considered in those patients with continuous air extravasation unless a cuffed tube can be placed distal to the site of injury. For children in whom airway injury occurs in a previously operated area, the risk of extravasation is reduced. This risk is also diminished if positive pressure ventilation can be avoided or minimized. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:921-924, 2021.

The impact of persistent pediatric obstructive sleep apnea on the Quality of Life of Patients' families.

OBJECTIVES: To (1) quantify the quality of life (QOL) for families of children affected by persistent obstructive sleep apnea (OSA), and (2) assess factors contributing to the negative impact of OSA on families. METHODS: Prospective case series in a multidisciplinary upper airway center at a tertiary pediatric institution. Our study included patients with persistent OSA referred to our clinic from 2014 to 2016. Both patients and their families completed validated questionnaires assessing QOL and OSA symptoms, including the Family Impact Questionnaire (FIQ), Pediatric Sleep Questionnaire, the Pediatric Quality of Life Inventory, the OSA-18, and the Epworth Sleepiness Scale. RESULTS: Families of 67 patients were included. The mean patient age was 12.5 (95%CI 11.9-13.1); 23 (34.3%) were female, and the mean BMI percentile was 80.2 (95%CI 73.6-86.8). The single most common comorbidity was Down syndrome (45.6%). The mean obstructive apnea-hypopnea index was 9.7 ±â€¯10.3 events/hour. A significant association exists between OSA severity and 18 FIQ negative subscore (P < 0.001). Financial impact was the primary negative concern for parents of patients with OSA compared to those without OSA (P = 0.03). There were no other significant differences between those with and without OSA. CONCLUSIONS: There was a significant correlation between persistent OSA severity and the FIQ negative impact of disease score on patients' families. Concern regarding financial burden was more common for families of children with OSA than for those without OSA. This suggests that targeted interventions, particularly regarding the financial burden of persistent OSA diagnosis and management, may provide some relief to families.

Pilot Study to Assess the Use of Ultrasound in Evaluating the Abnormal Pediatric Airway.

In this study, we sought to explore the feasibility of using ultrasonography to evaluate airway anomalies in awake children with previous airway reconstruction. For the month of December 2018, we reviewed the medical records of patients aged <18 years old with prior airway reconstruction who had an outpatient appointment and a microlaryngoscopy and bronchoscopy within 24 hours of each other. Four patients met inclusion criteria and were enrolled. Sonographic airway images and measurements were obtained during the outpatient appointment and compared with those obtained during endoscopy. Ultrasound identified extraluminal stents and glottic, subglottic, and tracheal pathology. Subglottic measurements obtained sonographically were within 0.1 to 0.5 mm of the outer diameter of the appropriate endotracheal tubes. Ultrasound did not visualize tracheotomy tubes or posterolateral pathology. Our findings lay the foundation for expanding the role of ultrasound in pediatric airway assessment, keeping in mind its apparent inability to visualize posterolateral airway pathology.

Timing of Initial Posttracheostomy Surveillance Endoscopy in Pediatric Patients.

OBJECTIVE: To determine the optimal timing of the first posttracheostomy microlaryngoscopy and bronchoscopy (MLB). STUDY DESIGN: Case series with chart review. SETTING: Tertiary pediatric medical center. SUBJECTS AND METHODS: Patients (<21 years of age) who underwent tracheostomy placement from January 1, 2011, to December 31, 2016. Patients were divided into early and late posttracheostomy surveillance groups (<6 weeks vs 6 to 14 weeks, respectively) based on the timing of their first posttracheostomy MLB. The primary outcome was to ascertain the clinical yield of the initial posttracheostomy MLB by documenting whether a medical or surgical treatment decision was made based on MLB findings. RESULTS: In total, 202 patients were included; of these patients, 162 met criteria for placement in the early group and 40 met criteria for the late group. There was no significant difference between the early and late groups regarding whether a medical or surgical decision was made at the time of the first MLB (21.5% vs 19%, respectively; P = .49). Multiple logistic regression identified that the presence of tracheostomy-related symptoms prior to MLB (odds ratio, 6.75; 95% confidence interval, 2.78-16.39) was the sole predictor of a medical or surgical decision being made at the first posttracheostomy MLB. CONCLUSION: The presence of tracheostomy-related symptoms was predictive of a medical or surgical decision being made using information obtained at the time of the first posttracheostomy MLB. We thus recommend that surveillance endoscopy be initiated when tracheotomized children start to develop tracheostomy-related symptoms.

A review of the effects of sleep during the first year of life on cognitive, psychomotor, and temperament development.

During the first year of life, infants spend most of their time in the sleeping state. Assessment of sleep during infancy presents an opportunity to study the impact of sleep on the maturation of the central nervous system (CNS), overall functioning, and future cognitive, psychomotor, and temperament development. To assess what is currently known regarding sleep during infancy and its effects on cognitive, psychomotor, and temperament development, we assessed the relevant literature published over the last several decades. To provide a foundation for a more in-depth understanding of this literature, we preface this with an overview of brain maturation, sleep development, and various assessment tools of both sleep and development during this unique period. At present, we do not have sufficient data to conclude that a causal relationship exists between infant sleep and cognitive, psychomotor, and temperament development. Caution should be used in predicting outcomes, as the timing and subjectivity of evaluations may obviate accurate assessment. Collectively, studies assess a wide array of sleep measures, and findings from one developmental period cannot be generalized readily to other developmental periods. Future studies should follow patients longitudinally. Additionally, refinements of existing assessment tools would be useful. In view of the relatively high reported pediatric prevalence of cognitive and behavioral deficits that carry significant long-term costs to individuals and society, early screening of sleep-related issues may be a useful tool to guide targeted prevention and early intervention.

Determinants of regional cerebral oxygenation in children with sleep-disordered breathing.

RATIONALE: An association between neurocognitive deficits and pediatric sleep-disordered breathing has been suggested; however, weak correlations between disease severity and functional outcomes underscore the lack of knowledge regarding factors modulating cognitive morbidity of sleep-disordered breathing. OBJECTIVES: To identify the parameters affected by sleep-disordered breathing that modulate cerebral oxygenation, an important determinant of cognition. A further objective was to use these parameters with demographic data to develop a predictive statistical model of pediatric cerebral oxygenation. METHODS: Ninety-two children (14 control subjects, 32 with primary snoring, and 46 with obstructive sleep apnea) underwent polysomnography with continuous monitoring of cerebral oxygenation and blood pressure. Analysis of covariance was used to relate the blood pressure, sleep diagnostic parameters, and demographic characteristics to regional cerebral oxygenation. MEASUREMENTS AND MAIN RESULTS: To account for anatomic variability, an index of cerebral oxygenation during sleep was derived by referencing the measurement obtained during sleep to that obtained during wakefulness. In a repeated measures model predicting the index of cerebral oxygenation, mean arterial pressure, rapid eye movement (REM) sleep, female sex, age, and oxygen saturation had a positive effect on cerebral oxygenation levels, whereas arousal index and non-REM (NREM) sleep had a negative effect. CONCLUSIONS: Increasing mean arterial pressure, age, oxygen saturation, and REM sleep augment cerebral oxygenation, while sleep-disordered breathing, male sex, arousal index, and NREM sleep diminish it. The proposed model may explain the sources of variability in cognitive function of children with sleep-disordered breathing.

Hearing loss progression and contralateral involvement in children with unilateral sensorineural hearing loss.

OBJECTIVES: We undertook this study to determine the rate of hearing loss progression in the affected ear of children with unilateral sensorineural hearing loss and without an enlarged vestibular aqueduct, and the rate of new-onset hearing loss in the contralateral ear. METHODS: We searched the database at our pediatric tertiary care center to identify patients who met the inclusion criteria, examining demographic variables, audiometric data, and presumptive causes. RESULTS: We identified 198 patients. At presentation, they showed slight left-sided and male predominances. Of 142 patients who had sufficient audiometric follow-up for us to evaluate progression, 21% showed ipsilateral progression and 10.6% developed new-onset hearing loss in the contralateral ear. Isolated high-frequency loss was identified in 11 patients (5.6%), 8 of whom had sufficient follow-up for us to identify progression. Two showed progression; 4 others with progression in the ipsilateral ear developed new-onset high-frequency loss in the contralateral ear. Temporal bone anomalies were identified in 26 children (13%), and these children were more likely to have profound hearing loss than were those without temporal bone anomalies (46% versus 23%). CONCLUSIONS: The findings suggest that unilateral sensorineural hearing loss may not always be a unilateral process, but that it may be the initial manifestation of bilateral auditory dysfunction.

The Effect of Cleft Palate Repair on Polysomnography Results.

STUDY OBJECTIVES: In view of the risk that surgical repair of cleft palate may induce or worsen obstructive sleep apnea (OSA), the goal of this study was to assess presurgical and postsurgical polysomnography (PSG) results for children who underwent primary palatoplasty. METHODS: Retrospective case-control series for children with cleft palate repair performed between January 2008 and December 2016 at a tertiary pediatric center. Children underwent PSG before and after surgery. RESULTS: Sixty-four children (53.1% female) with a mean age of 2.0 ± 2.8 years (range 0.6-16.4) were included in the study. Pierre-Robin sequence was the most common comorbidity (67%). Before palatal repair, the mean obstructive apnea-hypopnea index (oAHI) was 3.4 ± 3.9 (range 0-17.9) events/h; this did not significantly change, with 5.9 ± 14.5 (range 0-105.7) events/h after surgery (P = 0.30). However, 34.4% of patients had a worsening of more than 1 obstructive event/h and 18.9% had a worsening of 5 or more obstructive events/h. The presence of a concomitant syndrome (eg, Treacher Collins) was a risk factor for postoperative OSA (odds ratio 4.2, 95% confidence interval 1.1-15.8, P = .03). CONCLUSIONS: OSA did not develop or worsen following primary palatoplasty. However, the oAHI increased by 5 or more events/h in approximately 20% of study participants. The presence of a syndrome was the only factor predictive of worsening OSA after palatoplasty. These findings suggest that palatoplasty does not worsen or cause OSA in most patients, and that nonsyndromic children are at low risk for the development or worsening of OSA.

Impact of Balloon Diameter on Dilation Outcomes in a Model of Rabbit Subglottic Stenosis.

OBJECTIVE: To determine the appropriate balloon size for dilation using a previously described reproducible survival animal model of subglottic stenosis. STUDY DESIGN: Prospective animal study. METHODS: We conducted a prospective study including 16 New Zealand White rabbits. The airway of each animal was sized with an endotracheal tube (ETT), and subglottic stenosis (SGS) was endoscopically induced using Bugbee electrocautery to 75% of the circumference of the subglottis, followed by 4-hour intubation. Two weeks postoperatively, the rabbits' airways were sized and then dilated using a 6-, 7-, 8-, or 9-mm balloon, with four animals in each experimental group. Following dilation, animals were again sized and subsequently euthanized. The cricoid lumen was measured microscopically in each animal. RESULTS: Prior to inducing stenosis, all animals were sized with a 3.5 ETT. After inducing injury but prior to dilation, airways showed grade 2 SGS that sized with a 2.5 ETT with no leak. Postdilation, animals dilated with 6- or 7-mm balloons (n = 8) sized with a 3.0 ETT, and animals dilated with an 8- or 9-mm balloon (n = 8) sized with a 3.5 ETT. Postdilation median cricoid lumen measurements were 12.5 mm2 (6-mm balloon), 13.92 mm2 (7 mm), 16.83 mm2 (8 mm), and 17.15 mm2 (9 mm); two cricoid fractures occurred in the 9-mm group. CONCLUSION: The postdilation cricoid lumen diameter increased with increased balloon size, and the use of an 8-mm balloon achieved the largest cricoid lumen diameter without causing fracture. Further research is necessary to determine the ideal duration of dilation and optimal intervals between dilations. LEVEL OF EVIDENCE: NA Laryngoscope, 129:2409-2413, 2019.

Endoscopic posterior cricoid reduction: A surgical method to improve posterior glottic diastasis.

OBJECTIVES/HYPOTHESIS: Describe the preoperative evaluation of patients with glottic diastasis who are candidates for endoscopic posterior cricoid reduction (EPCR) and their perioperative and postoperative surgical and voice outcomes, and validate the aerodynamic benefit of EPCR using computation fluid dynamics (CFD)-based modeling from computed tomography (CT) scans. STUDY DESIGN: Retrospective case series. METHODS: Thirteen patients who underwent EPCR were followed from 2013 to 2017. They received a preoperative voice evaluation, microlaryngoscopy and bronchoscopy, dynamic voice CT (performed on patients seen from 2014 to study completion), and postoperative voice evaluation (n = 12). Postoperative inpatient days, complications, and postoperative endoscopic intervention were collected. To validate the aerodynamic benefit of EPCR, CFD modeling was carried out on one patient. RESULTS: Thirteen patients (nine females, nine with intubation injury, and four with post-airway reconstruction dysphonia) underwent EPCR at a mean age of 11.0 years. The mean preoperative and postoperative Pediatric Voice handicap Index scores were 53.8 and 33.8, respectively (P = .006). Mean maximum phonation time preoperatively and postoperatively was 5.3 and 6.7, respectively (P = .04). Of eight patients who underwent preoperative CT imaging, all demonstrated a posterior gap. Modeling demonstrated a change in flow and pressure. The mean hospital stay was 2.4 days. Nine patients underwent postoperative endoscopic intervention, and one experienced a complication that resolved with intervention. CONCLUSIONS: Patients who underwent EPCR for dysphonia following intubation or airway reconstruction showed improvements in vocal efficiency, loudness, and perceived voice handicapping. Their hospital stay was brief, with few complications. CFD modeling corroborated these clinical findings. EPCR thus warrants consideration in the management of patients with posterior glottic diastasis. LEVEL OF EVIDENCE: 4 Laryngoscope, 129:S1-S9, 2019.

Development of a survival animal model for subglottic stenosis.

OBJECTIVE: To develop a reproducible survival animal model for subglottic stenosis. STUDY DESIGN: Prospective study. METHODS: We evaluated five methods of inducing airway injury in 30 New Zealand white rabbits to produce a subglottic stenosis model. Experimental groups comprised: group 1 (n = 5), which underwent 4-hour intubation; group 2 (n = 5), which underwent induced subglottic injury with a nylon brush; group 3 (n = 10), which underwent subglottic injury with a nylon brush, followed by 4-hour intubation; group 4 (n = 5), which underwent subglottic injury with Bugbee cautery in 50% of the subglottic circumference, followed by 4-hour intubation; and group 5 (n = 5), which underwent subglottic injury with Bugbee cautery in 75% of the subglottic circumference, followed by 4-hour intubation. Five animals were used as controls. Endoscopy of the airway and sacrifice of animals were planned at an interval of 14 days postinjury. Histologic measurements were analyzed. RESULTS: No animals in groups 1 or 2 developed stenosis. In group 3, 50% of animals developed symptomatic grade 3 subglottic and tracheal stenosis, necessitating early endoscopy and sacrifice in three animals. Four animals in group 4 developed grade 1 subglottic stenosis, and four in group 5 developed grade 2 subglottic stenosis. Histologic measurements of lumen areas within each of these two groups were similar; all animals survived the follow-up period. CONCLUSION: We successfully developed a reproducible survival model for induced subglottic stenosis using a combination of cautery-induced subglottic injury followed by 4-hour intubation. This model lays the foundation for future studies that evaluate endoscopic interventions for the management of subglottic stenosis. LEVEL OF EVIDENCE: NA Laryngoscope, 129:989-994, 2019.

Comparison of Patient- and Parent-Reported Quality of Life for Patients Treated for Persistent Obstructive Sleep Apnea.

Objectives To evaluate the impact of the treatment of persistent pediatric obstructive sleep apnea (OSA) on quality of life (QOL) with patient-reported outcomes tools and to compare parent- and self-reported Pediatric Quality of Life Inventory (PedsQL) scores. Study Design Prospective case series. Setting Multidisciplinary upper airway center at a tertiary pediatric institution. Subjects and Methods Children with persistent OSA referred to our multidisciplinary upper airway center from 2014 to 2016. Patients and their families completed validated questionnaires for QOL, including the Family Impact Questionnaire, the Obstructive Sleep Apnea-18, the PedsQL, and the Epworth Sleepiness Scale for Children and Adolescents. They completed the same surveys after treatment. Results Twenty-three children (7 females) and their families were included in the study. Patients had a mean age of 12.7 years. Pretreatment, the mean obstructive apnea-hypopnea index was 15.0 events/hour (95% CI, 8.7-21.3); after treatment, the mean was 3.9 events/hour ( P = .003). The mean PedsQL score for children improved from 60.5 to 74.2 ( P = .04). The PedsQL score for parents did not significantly improve (61.6 to 63.8, P = .39). There was a significant though weak association between OSA severity ( R2 = 0.25, P = .03) and PedsQL scores. The Family Impact Questionnaire negative subscore and Epworth Sleepiness Scale for Children and Adolescents scores did not change with treatment. Conclusions For children treated for persistent OSA, we found that self-reported QOL significantly improved after treatment; however, parent-reported QOL did not significantly change. It is unclear if parents underestimate or patients overestimate QOL after treatment. We suggest that patient-reported outcomes be obtained when feasible.

A shared decision-making tool for obstructive sleep apnea without tonsillar hypertrophy: A randomized controlled trial.

OBJECTIVES/HYPOTHESIS: Shared decision-making is a process whereby patients and clinicians jointly establish a treatment plan integrating clinical evidence and patient values and preferences. Although this approach has been successfully employed in numerous medical disciplines, often using shared decision-making tools, otolaryngologic research assessing its use is scant. Our primary objective was therefore to determine if the tools we developed reduced decisional conflict for children with obstructive sleep apnea without tonsillar hypertrophy. STUDY DESIGN: Prospective, single-blind, randomized controlled trial. METHODS: We enrolled consecutive patients meeting inclusion criteria who were referred to our multidisciplinary upper airway center. Study patients used a shared decision-making tool whereas controls did not. Measures of decisional conflict (SURE [Sure of myself, Understanding information, Risk benefit ratio, Encouragement], CollaboRATE, and the Decisional Conflict Scale [DCS]) were obtained pre- and postvisit. RESULTS: We assessed 50 families (study group = 24, controls = 26). The mean age was 8.8 ± 6.6 years, 44% were female, 86% were white, and the mean obstructive apnea-hypopnea index was 12.7 ± 15.6 events/hour. The previsit mean DCS score was similar for controls (42.7) and study patients (40.8) (P = .38). The postvisit mean DCS score for controls was 13.3 and for study patients 6.1 (P = .034). Improvement in this score was greater in the study group (P = .03). At previsit evaluation, 63% of controls and 58% of study patients were unsure about their options. Postvisit, this improved to 4.1% and 0%, respectively. CONCLUSIONS: Families counseled regarding treatment options using shared decision-making tools had significantly less decisional conflict than those who did not use these tools. These positive outcomes suggest that clinicians should consider integrating this approach into clinical practice. LEVEL OF EVIDENCE: 1b. Laryngoscope, 128:1007-1015, 2018.

Characterizing Decisional Conflict for Caregivers of Children With Obstructive Sleep Apnea Without Tonsillar Hypertrophy.

STUDY OBJECTIVES: The goals of this study were to (1) evaluate the degree of decisional conflict (DC) experienced by caregivers of children with obstructive sleep apnea (OSA) without tonsillar hypertrophy; and (2) describe the association between DC, quality of life (QOL), and OSA severity. METHODS: This study comprised children evaluated in the multidisciplinary upper airway center at the Cincinnati Children's Hospital Medical Center from December 2014 to May 2016. Caregivers were asked to complete surveys (Pediatric Quality of Life Inventory 4.0 [PedsQL], OSA-18, Epworth Sleepiness Scale, Family Impact Questionnaire, Decisional Conflict Scale, CollaboRATE scale, and SURE questionnaire) during a clinic visit. Polysomnography data were collected. Analysis included Kruskal-Wallis, Wilcoxon rank-sum, and regression testing. RESULTS: Caregivers of 76 children participated; 16 (21.1%) had high DC. There were no significant differences in demographics between those with low and high DC; the low DC group had a higher obstructive apnea-hypopnea index (13.2 versus 12.3 events/h; P = .013). Overall and disease-specific QOL, sleepiness, family impact scores, and DC did not differ by OSA severity except for the PedsQL physical subcategory (P = .02). DC was associated with the total PedsQL (P = .043) on univariate regression; however, this did not persist (P = .61) after controlling for demographic variables. DC scores correlated well with CollaboRATE and SURE throughout the analysis (P < .001). CONCLUSIONS: The proportion of caregivers of children with OSA without tonsillar hypertrophy who experienced a high level of DC regarding their child's treatment was 21.1%. Neither DC nor OSA severity was related to QOL in children with OSA. The briefer SURE or CollaboRATE scales were adequate tools to measure DC in these children.

Circadian Dysregulation: The Next Frontier in Obstructive Sleep Apnea Research.

OBJECTIVE: To review the effects of the circadian clock on homeostasis, the functional interaction between the circadian clock and hypoxia-inducible factors, and the role of circadian dysregulation in the progression of cardiopulmonary disease in obstructive sleep apnea (OSA). DATA SOURCES: The MEDLINE database was accessed through PubMed. REVIEW METHODS: A general review is presented on molecular pathways disrupted in OSA, circadian rhythms and the role of the circadian clock, hypoxia signaling, crosstalk between the circadian and hypoxia systems, the role of the circadian clock in cardiovascular disease, and implications for practice. Studies included in this State of the Art Review demonstrate the potential contribution of the circadian clock and hypoxia in animal models or human disease. CONCLUSIONS: Molecular crosstalk between the circadian clock and hypoxia-inducible factors has not been evaluated in disease models of OSA. IMPLICATIONS FOR PRACTICE: Pediatric OSA is highly prevalent and, if left untreated, may lead to cardiopulmonary sequelae. Changes in inflammatory markers that normally demonstrate circadian rhythmicity are also seen among patients with OSA. Hypoxia-inducible transcription factors interact with core circadian clock transcription factors; however, the interplay between these pathways has not been elucidated in the cardiopulmonary system. This gap in knowledge hinders our ability to identify potential biomarkers of OSA and develop alternative therapeutic strategies. A deeper understanding of the mechanisms by which OSA impinges on clock function and the impact of clock dysregulation on the cardiopulmonary system may lead to future advancements for the care of patients with OSA. The aim of this review is to shed light on this important clinical topic.

The Management of Cyanotic Spells in Children with Oesophageal Atresia.

Cyanotic spells, also known as blue spells, dying spells, or apparent life-threatening events, refer to a bluish tone visible in the mucosal membranes and skin caused by an oxygen decrease in the peripheral circulation. Although this decrease may be transient and benign, it may also be indicative of a severe underlying problem that requires immediate intervention. Children with oesophageal atresia (OA) are at risk for a number of coexisting conditions that may trigger cyanotic spells. This current article will focus on the management of cyanotic spells both in children with innominate artery compression and those with tracheomalacia.

Temporal trends in inpatient pediatric sleep apnea surgery: 1993-2010.

OBJECTIVES/HYPOTHESIS: We sought to characterize changes in the patterns of inpatient surgical sleep care over time and ascertain if these changes were consistent with previously reported trends in adult surgical sleep care. STUDY DESIGN: Repeated cross-sectional study. METHODS: Discharge data from the U.S. Nationwide Inpatient Sample for 125,691 nasal, palatal, or hypopharyngeal procedures in children for sleep-disordered breathing or obstructive sleep apnea (OSA) from 1993 to 2010 were analyzed using cross-tabulations and multivariate regression modeling. RESULTS: Inpatient surgical sleep procedures increased from 45,671 performed in 1993 to 2000 (study period 1) to 80,020 in 2001 to 2010 (study period 2). Overall, patients were commonly male (61.3%), privately insured (46.8%), 2 to 6 years old (39.4%), and white (36.4%). Obesity was reported in 4.6% and 6.7% of children during study periods 1 and 2, respectively (P < .0001). Tonsillectomy (with and without adenoidectomy) was the most commonly performed procedure in both study periods. With the exception of uvulopalatopharyngoplasty and tracheostomy, all sleep procedures increased over time; however, multilevel surgery did not significantly increase (P = .28). Children who underwent sleep surgery during study period 2 were more likely to receive a supraglottoplasty (P = .0125) and to undergo procedures at high-volume hospitals (P = .0311), and less likely to undergo a tracheostomy (P < .0001). CONCLUSIONS: These data reflect changing trends in the surgical management of pediatric OSA, with significant increases in nasal and hypopharyngeal procedures, particularly lingual tonsillectomy and supraglottoplasty. Unlike the trend in adults, multilevel surgery in children with sleep disordered breathing or OSA has not yet been integrated into routine clinical practice. LEVEL OF EVIDENCE: 2C Laryngoscope, 127:1235-1241, 2017.

Deficient tracheal rings.

Deficient cartilaginous rings are an extremely rare intrinsic tracheal defect. To our knowledge, only two cases have previously been reported in the literature. We describe our experience with three patients presenting with this unusual defect causing severe tracheomalacia who were successfully managed with slide-tracheoplasty. With respect to correction of the stenotic tracheal segment, our clinical outcomes were excellent. Dissimilarities in medical history suggest that the origin of deficient tracheal rings can be either congenital or acquired.

Subglottic stenosis.

Subglottic stenosis (SGS) is a congenital or acquired condition characterized by a narrowing of the upper airway extending from just below the vocal folds to the lower border of the cricoid cartilage. With the introduction of prolonged intubation in neonates (mid 1960s), acquired SGS became the most frequent cause of laryngeal stenosis; unlike congenital SGS, it does not improve with time. Laryngeal reconstruction surgery evolved as a consequence of the need to manage these otherwise healthy but tracheotomized children. Ongoing innovations in neonatal care have gradually led to the salvage of premature and medically fragile infants in whom laryngeal pathology is often more severe, and in whom stenosis often involves not only the subglottis, but also the supraglottis or glottis-causing significant morbidity and mortality. The primary objective of intervention in these children is decannulation or preventing the need for tracheotomy. The aim of this article is to present a more detailed description of both congenital and acquired SGS, highlighting the essentials of diagnostic assessment and familiarizing the reader with contemporary management approaches.