bluuebunny/crossref_metadata_2025_split huggingface dataset:
0779 Home Sleep Apnea Test for Monitoring Pediatric Obstructive Sleep Apnea Following Management with Adenotonsillectomy
Authors: Roman-Rosado Yaelis , Cielo Christopher , Tapia Ignacio , Lin Nancy , Stefanovski Darko , Ward Michelle , Ziccardi Lois 
Published: April 2024

Abstract

Introduction
Many children have residual obstructive sleep apnea (OSA) following adenotonsillectomy (AT). While in-lab polysomnography (PSG) remains the gold standard for re-evaluation, its limitations include cost, limited availability of pediatric sleep laboratories, and inconvenience to families due to disruptions in routine. Home sleep apnea testing (HSAT), used clinically in adults, presents an alternative to evaluate the effect of OSA treatment in children and improve access to care. Specifically, type II HSAT, may surpass limited-channel tests in accuracy. This study aims to evaluate the feasibility, acceptability, and preference of type II HSAT compared to PSG to evaluate treatment effect following AT in children with OSA.

Methods
This comparative effectiveness trial included children aged 5-12 years scheduled for a PSG to evaluate residual OSA following AT. Participants underwent type II HSAT in their home within one week of their clinical PSG. Children and parents completed questionnaires regarding preference and acceptability of type II HSAT versus PSG. The relationship between obstructive apnea-hypopnea index (OAHI) by HSAT and PSG was analyzed using Spearman correlation and Bland-Altman plots.

Results
In this preliminary analysis, 20 participants (10 female; mean age=9.5; Black/African American 70%; White 25%; Hispanic 10%) who attempted HSAT completed all study procedures. Thirteen (65%) parents reported HSAT was closer to a normal night’s sleep compared to PSG, and 11 (55%) preferred PSG. Using an OAHI cutoff of 2 events/hour, OSA status by HSAT was the same as PSG for 16/20 (80%) participants. Spearman’s correlation was 0.8 (p-value &lt; 0.001). The Bland-Altman analysis revealed the average difference (-1.025), falling within the 95% limits of agreement (-4.694, 2.644).

Conclusion
Based on preliminary analysis, parents reported type II HSAT was feasible and closer to a normal night’s sleep compared to PSG. Type II HSAT was accurate in detecting mild OSA and importantly, HSAT-derived OAHI correlated well with PSG. To further validate these initial findings, we aim to enroll 40 more patients in this ongoing trial. Type II HSAT appears to be acceptable and have comparable accuracy to be considered an alternative to PSG to re-evaluate OSA following AT in school-age children.

Support (if any)
This research is supported by the AASM Foundation.
0782 Persistent Obstructive Sleep Apnea in Medically Complex Pediatric Patients: A Multidisciplinary Treatment Approach
Authors: Petitto Lacie , Revana Amee , Hays Brooke , Tran Elly , Kazmerski Jennifer , Virani Farrukh , Musso Mary 
Published: April 2024

Abstract

Introduction
Treatment of persistent obstructive sleep apnea (OSA) in medically complex pediatric patients can be challenging due to concurrent developmental delays contributing to poor adherence with positive airway pressure (PAP) therapy. There is limited literature describing a multidisciplinary approach to treating persistent OSA in medically complex pediatric patients utilizing a pulmonologist, otolaryngologist, sleep medicine provider, psychologist, nutritionist, and nurse coordinator. Objectives: 1: Describe the multidisciplinary approach to treating persistent OSA in children at our institution; 2: Review concurrent patient diagnoses and interventions recommended; 3: Report resulting decreased patient costs and increased access to care.

Methods
We developed a multidisciplinary OSA (MOSA) clinic for patients with recalcitrant OSA in November 2022 in a pediatric tertiary care setting. Patients with persistent OSA after adenotonsillectomy who failed PAP therapy were seen for treatment optimization. This included consideration for drug induced sleep endoscopy (DISE) and/or Hypoglossal Nerve Stimulator (HGNS), as well as an evaluation by sleep psychology, pulmonary medicine, and a sleep provider to improve PAP adherence. Data was collected and reviewed via retrospective chart review.

Results
Twelve clinic sessions have been completed and 46 pediatric patients have been evaluated (n=46) with an average age of 10.4 years (±5 years). 58% were referred for nonadherence to PAP therapy. The most prevalent concurrent diagnosis was developmental delay (n=36, 78%). Other diagnoses included Trisomy 21 (n=18, 46%), other genetic disorders (n=8, 17%), and autism (n=6, 13%). 61% (n=46) had &gt;95th%ile BMI-for-age resulting in nutrition consultation. Surgical intervention was recommended for 41% (n=19) while 43% (n=20) were scheduled for ongoing desensitization to PAP therapy. Combining specialties reduced average patient travel by 220 miles and gas costs of $35.44. Wait times to see the otolaryngologist decreased from 9 months to 3 months. Due to high demand, a second clinic location will launch in February 2024.

Conclusion
Our MOSA clinic has provided significant value in treating complex pediatric patients with persistent OSA. This review identifies patients with comorbidities that may be good candidates for a multidisciplinary sleep clinic, the potential improvement in access to care, and cost savings for patients and families requiring complex care related to persistent OSA.

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0784 Spinal Muscular Atrophy 1 Genetic Therapies Halt Development of Infantile Onset Sleep-Related Disordered Breathing
Authors: Suri Christian , Palladino Christine , Hogan Katherine , Laureta Emma , Irumudomon Obehioya , Rajan Saji , Kelly-Pieper Kristin , Kothare Sanjeev 
Published: April 2024

Abstract

Introduction
Natural history studies of Spinal Muscular Atrophy Type 1 demonstrate that patients typically require non-invasive ventilatory interventions in the first year of life for sleep-related disordered breathing (e.g. obstructive sleep apnea, central sleep apnea, and hypoventilation) due to the progressive development of neuromuscular weakness. There are currently no reports that analyze the effects that disease-modifying therapy has on the development of sleep-related disordered breathing.

Methods
This descriptive retrospective cohort study will utilize a Kaplan-Meier time-to-event analysis to determine whether administration of disease-modifying therapy delays diagnosis of sleep-related disordered breathing and halts initiation of ventilatory assistance in the first year of life.

Results
Six patients with spinal muscular atrophy type 1 followed by the division of pediatric neurology were included in the cohort. One patient received both nusinersen and onasemnogene abeparvovec in the first year of life and two patients received onasemnogene abeparvovec in the first year of life followed by risdiplam after one year of life. Three patients exclusively received onasemnogene abeparvovec in the first year of life. Four of the patients did not receive a sleep study, as respiratory pathology was not a concern at follow up visits due to preservation or improvement of motor strength. The two that received polysomnograms were diagnosed with obstructive sleep apnea after the first year of life with an average AHI of 4.1, average SpO2 nadir of 81.5%, and average total arousal index of 10.

Conclusion
Though a small sample size, there is some evidence to suggest that prompt initiation of disease-modifying therapy such as nusinersen, onasemnogene abeparvovec, ,and risdiplam can possibly halt the development of sleep-related disordered breathing in the first year of life and delay initiation of ventilatory support. Further controlled trials would be needed to further understand the relationship between disease-modifying therapy and sleep-related respiratory outcomes in pediatric patients with SMA type 1.

Support (if any)
0785 Effects of Sympathetic Activity on Energy Intake in Upper Airway Obstruction/ Obstruction Removal Mouse Model
Authors: Nujedat Haiat , Arazi Eden , Assadi Mohammad H , Segev Yael , Tarasiuk Ariel 
Published: April 2024

Abstract

Introduction
Pediatric sleep-disordered breathing (SDB) increases the risk of accelerated body weight gain if the post-treatment weight is uncontrolled. Upper airway obstruction (AO)/ obstruction removal (OR) in rodents mimics many SDB characteristics, including increased food intake. Orexin, a peptide produced by hypothalamic neurons, emerged as one of the critical orchestrators of breathing and deregulation of feeding in AO/ OR animals. It is possible that orexin affects energy expenditure due to increased sympathetic activation of brown fat uncoupling protein-1 (UCP-1) that may contribute to weight gain following OR. We hypothesize that AO/OR induces elevation of sympathetic activity that leads to persistent increased feeding behavior.

Methods
We employed a mouse model to examine the impact of AO followed by OR two weeks later. The mice were observed for an additional eight to twelve weeks, roughly equivalent to two decades in human terms. We examined the impact of AO/OR on feeding hormones, respiration (assessed through plethysmography), energy expenditure and intake (monitored in metabolic cages), nutritional preferences for a high glycemic index diet, and thermography using infrared cameras. We studied the impact of specific β3-adrenergic receptor blockade (L-748,337, 5 mg/kg) on energy balance.

Results
The heightened energy expenditure in AO was associated with an augmented breathing workload, elevated plasma norepinephrine levels, increased UCP1 levels, and an enhanced effort in breathing to maintain acid-base homeostasis. Elevated orexin and ghrelin levels were associated with increased energy intake. The AO mice's daily dietary glycemic index intake exceeded that of the control group by more than 250% compared to a low glycemic index diet. OR maintained heightened food intake, preference for a high glycemic index diet, and elevated energy expenditure despite normalized ventilation, associated with increased interscapular temperatures and brown fat UCP1 levels. The administration of L-748,337 reduces interscapular temperatures and decreases energy intake in the OR group.

Conclusion
The need to maintain respiratory homeostasis in AO animals is associated with abnormal energy metabolism and thermogenic capacity that are not reversible following OR. Obstruction removal is linked to persistent heightened feeding and increased sympathetic activity, even with normalized breathing and energy needs.

Support (if any)
Israel Science Foundation Grant No. 164/2018.
0786 Associations of Physiological Traits with Pediatric Sleep Apnea Severity: Variation by Race and Ethnicity
Authors: Tsou Po-Yang , Alex Raichel , Redline Susan , Sands Scott 
Published: April 2024

Abstract

Introduction
Adenotonsillar hypertrophy increases risk for pediatric obstructive sleep apnea (OSA), likely due to increased upper airway collapsibility. Pathophysiologic determinants for OSA (or endotypes) included greater pharyngeal collapsibility, reduced dilator muscle compensation, elevated chemoreflex loop gain, and altered arousal threshold. More severe OSA and higher prevalence of residual OSA after adenotonsillectomy in Black compared to White children suggest differences in endotypes. We aimed to determine the extent to which changes in endotypes vary with participant characteristics and may explain differences in OSA severity across groups.

Methods
Endotypes were estimated during REM- and NREM- sleep from baseline polysomnography in children with OSA screened for participation in the Childhood Adenotonsillectomy Trial (CHAT; N=1232; age:3-9.9(y); BMI(z):0.88±1.24 [mean±SD]; Black: 47.9%; Asian:2.1%; Female:52.8%). Race and ethnicity differences in REM- and NREM-AHI levels and endotypes were examined adjusting for age, sex, BMI(z), and time in lateral position. Multivariable regression models assessed associations of REM- and NREM-endotypes (per SD) with REM- and NREM-apnea-hypopnea index (AHI; events/h), mutually adjusted for other endotypes. Mediation analysis quantified the extent to which endotypic differences explained race differences in AHI.

Results
The sample had a baseline AHI:4.9±8.9; REM-AHI:10.6±19.7; NREM-AHI:3.6±7.6 (mean±SD). In sex, age and BMI-adjusted multivariable analyses, AHI was higher in Black (REM-AHI: 7.81±1.01 events/h; NREM-AHI: 1.57±0.30 events/h; β±SEM) and Asian (REM-AHI: 9.37±3.35 events/h) children compared to White children. Compared with White children, Black children had higher collapsibility (0.30±0.09; β±SEM) and higher arousal threshold (0.21±0.11) in REM, while Asian children had decreased compensation (REM: −1.06±0.22; NREM: −0.48±0.11) in REM and NREM. Sex or BMI(z) were not associated with collapsibility or compensation. Endotypic analysis, adjusted for lateral position only, showed that higher REM- and NREM-AHI were associated with greater collapsibility (REM: 13.64±1.73; NREM: 5.58±0.57; β±SEM), reduced compensation (REM: −4.22±0.98; NREM: −1.88±0.23) and higher arousal threshold (REM: 3.42±0.97; NREM: 0.72±0.17). Mediation analysis showed that the higher REM-AHI in Black children was partially explained by elevated arousal threshold (0.21 SD; percentage mediated=19 [95%CI:3-39]%).

Conclusion
Endotypes are associated with pediatric OSA severity, and partially explain race-associated differences in AHI. These results suggest opportunities for targeting mechanistic traits to improve the outcomes of surgical therapies for OSA.

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0787 Clinical Observations and Characteristics of a Pediatric Cohort with OSA Treated with PAP for over 10 Years
Authors: Scribner April , Jambhekar Supriya 
Published: April 2024

Abstract

Introduction
Despite intensive treatment and follow-up, adherence to positive airway pressure in children is poor, with many losing the device or simply being lost to follow-up. The purpose of this study was to describe the characteristics of and management interventions in a cohort of 28 patients with obstructive sleep apnea treated with positive airway pressure followed in a pediatric sleep center for 10 years or longer.

Methods
A retrospective chart review was performed on patients diagnosed with obstructive sleep apnea and treated with positive airway pressure that were followed in clinic between May 2007 and December 2023. Those that followed up for less than 10 years were excluded from the analysis. Demographic and clinical variables were reviewed. A descriptive statistical analysis was performed.

Results
In this cohort (85.7% male), the median age (interquartile range) at PAP initiation was 4 years (2-7). Comorbidities included 42.9% obese, 39.3% allergic rhinitis, 35.7% asthma, 28.6% intellectual disability, 17.9% ADHD, 17.9% Prader-Willi syndrome, 10.7% autism, and 10.7% Down syndrome. 92.9% received tonsillectomy, 85.7% adenoidectomy, 50% uvulapalatopharyngoplasty, and 10.7% turbinate trim. . Polysomonogram (PSG) results [median (IQR)] included apnea-hypopnea index 15.89 per hour (10.15-29.25), arousal index 15.7per hour (11.85-25.85), 77.8% had oxygenation defects, and 22.2% ventilation defects.67.9% (19) participated in an intensive adherence program, with 42.1% (8) graduating the program The most common interventions/recommendations were mask/tubing replacement (22.2%), PSG ordered (11.8%), pressure change (10.5%), and changing mask type (10.3%). Of those changing masks, 45.3% changed from nasal to full-face, 15.1% from full-face to hybrid, and 13.2% from hybrid to full-face. Median PAP adherence across all patients and visits was 94.15 (81.88-99) and usage for four hours or more was 77.9 (54.10-90.93).

Conclusion
This study provides a detailed overview of demographical and clinical characteristics of and the management interventions for OSA in a pediatric cohort followed for over a decade. Further statistical analyses including multivariate analyses are in progress.

Support (if any)
none
0789 Implementation of Auto CPAP Pathway to Reduce Therapy Wait Times for OSA Management in Children
Authors: Berichon Leah , Annis Karen , Imran Sehyr , Kalra Maninder 
Published: April 2024

Abstract

Introduction
Auto-titrating Continuous Positive Airway Pressure devices (auto CPAP) has been reported to be safe and well tolerated in children. This study evaluates the impact of the implementation of the auto CPAP pathway on therapy wait times for obstructive Sleep Apnea (OSA) management in children.

Methods
All children 2-18 years of age diagnosed with OSA at Nationwide Children’s Sleep Center and started on PAP therapy in 2023 were included in the study. Descriptive statistics for demographics and student’s t -test to compare the two groups for time to initiation of therapy were calculated using R open-source software version 4.3.2.

Results
Among study participants, 30 children (mean age 14.6 years; range 9.7 to 17.6 years) were set on auto-CPAP and 32 children (mean age 13.4 years; range 5.7 to 17.9 years) were set up on CPAP after completing titration study. The median number of days from diagnostic PSG to auto-CPAP set up was 39 days (range of 9 to 139 days), as compared to a median of 74 days (range of 5 to 544 days) from diagnostic PSG to CPAP set up after completion of a titration study; P value was 0.002. The median number of days from order placement to auto-CPAP set up was 23 days (range of 2 to 69 days). Compliance data at 90 days was available in 17 children in the auto-CPAP group and 29 children in the titration-study CPAP group. A trend for higher compliance in auto-CPAP group (47%) versus the titration-study CPAP group (34%) was noted (P value = 0.09).

Conclusion
Implementation of auto-titration CPAP pathway significantly reduces the time to therapy initiation in children with OSA and should be offered when clinically indicated. Our results also indicate that delay from order placement to auto-CPAP setup is a key driver to delay in therapy initiation.

Support (if any)
0791 Polysomnographic Outcomes of Obstructive Sleep Apnea in Infants with Pierre-Robin Sequence
Authors: Sivaram Ganeshraj , Jawad Kahir , El-Kersh Karim , Chariker Mark , Betz Kelly , Senthilvel Egambaram 
Published: April 2024

Abstract

Introduction
Children with Pierre-Robin sequence (PRS) are known to have a high association with sleep-disordered breathing (SDB). Mandibular distraction osteogenesis (MDO), a surgical process involving lengthening of the jaw, has been used to improve SDB in these patients, preventing the need for tracheostomy or positive airway pressure therapy. Currently, limited data regarding polysomnographic outcome of SDB after mandibular distraction is available. Therefore, we aimed to assess the prevalence of SDB in pediatric patients with PRS and the polysomnographic outcome of MDO.

Methods
This was a single-center retrospective study that included children with confirmed PRS who underwent polysomnography (PSG) for evaluation of SDB. Obstructive sleep apnea (OSA) severity was categorized by the obstructive apnea-hypopnea index (OAHI) as normal (&lt; 1 events/hr), mild (1–4.9 events/h), moderate (5–9.9 events/h), and severe (≥ 10 events/h). Pre- and post-MDO PSG variables were compared using a paired t-test.

Results
Baseline PSG data were available for 16 children (8 males, 8 females) with mean age 374.2 days. Average BMI was 14.4 (±2.4 kg/m3) and 87.5% of patients were Caucasian. OSA prevalence was 87.5%, apnea hypopnea index (AHI) was severe in 68.8% and the mean AHI was 46.3 (± 43.8). Eleven patients had MDO with a mean age of 27.1 days (± 20.5) at time of MDO and distraction duration of 33.2 days (± 7.6). Six patients had pre and post MD PSGs. Average total sleep time (TST) increased significantly from 207.3 min (± 85.7 min) to 351.3 min (± 126.7 min); p=0.023. Average apnea-hypopnea index (AHI) and average REM AHI decreased significantly from 79.0 events/h (± 41.6 events/h) to 18.2 events/h (± 25.0 events/h), p=0.017; and 106.3 events/h (± 43.4 events/h) to 37.0 events/h (± 51.0 events/h), p=0.040, respectively. Average oxygen saturation nadir during sleep significantly improved from 72.0% (± 5.1%) to 85.2% (± 12.4%), p=0.030; and percentage of carbon-dioxide time spent above 50 mmHg improved significantly from 68.0% (± 17.7%) to 49.8% (± 6.0%) p=0.027.

Conclusion
Children born with PRS have a high prevalence of severe OSA. Additionally, MDO significantly improves TST, AHI, oxygen saturation nadir, and CO2 levels in children with SDB and PRS.

Support (if any)
0792 Relationships Between Age, REM Sleep, and Sleep Disordered Breathing Across the First 24 Months of Life
Authors: Nakra Neal , Morphew Tricia , Sayrs Lois , Blaine Kevin , Galut Lia , Polk Spencer , Simon Katharine C 
Published: April 2024

Abstract

Introduction
Infant airway anatomy and immature central respiratory systems place infants at risk for sleep disordered breathing. Further, infant sleep is comprised of higher amounts of rapid eye movement (REM) sleep, with a steady decline across the first twenty-four months. Compared to the controlled breathing rate during non-REM (NREM), REM sleep has an erratic rate. The combination of immature anatomy and increased REM amount predispose infants to greater apnea risk, yet there is a poor characterization of age- and REM-associated changes in disordered breathing across the first twenty-four months.

Methods
We retrospectively analyzed 95 clinical sleep studies from 50 infants (M=10.5 months, SD = 7.6., F=24), each had one to four records, who were being evaluated for sleep disordered breathing. We investigated the average number of combined central and obstructive apnea events per hour, controlling for age of infant, and percentage of total sleep time in both NREM and REM, respectively, using a Poisson regression within the Generalized Linear Mixed Model (GLMM) framework.

Results
Infants slept on average 371 minutes (SD=121), of which approximately 33% (SD=12%) was spent in REM. We observed an average of 5.3 apnea events per hour (SD = 5), with a median of 3.5 [IQR: 1.7, 7.9], unadjusted, which we found varied by age (p&lt;.05). During the first year, higher apnea event rates were observed at one and six months, while the lowest apnea rate occurred at three months of age. Between 12 and 24 months, rates stabilized, mirroring those observed at three months, but with a noticeable increase at 18 months. There was a strong relationship with REM sleep, with 3.2 times more apnea events per hour during REM versus nREM. This increased rate during REM was consistent across age, including when adjusted for the percentage of REM sleep and sex (RR=3.18, 95% CI 2.61, 3.89, p&lt;.001).

Conclusion
We observed a dynamic relationship between age, REM sleep, and total apnea events across the first twenty-four months of life. As infants are at higher risk for apnea, evaluating age- and REM-associated apnea changes may provide new assessment and intervention targets in sleep disordered breathing.

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