bluuebunny/crossref_metadata_2025 huggingface dataset:
0771 Sex and Racial Disparities in Obstructive Sleep Apnea Among Adolescents
Authors: Gutierrez Jeremy Landeo , Ryu Julie , Tantisira Kelan , Bhattacharjee Rakesh
Published: April 2024

Introduction

Obstructive sleep apnea (OSA) gender dimorphism in the pediatric population has not been elucidated, while there has been a paucity in studies with adequate representation of adolescents. We hypothesize that OSA prevalence in adolescents undergoing polysomnography (PSG) is higher among male sex subjects than their female peers, even after adjusting for body mass index and other covariates.

Methods

Retrospective review of adolescents 11 to 18 years old who underwent a PSG at Rady Children’s Hospital between October 2016 and April 2022. Exclusion criteria: craniofacial and chromosomal anomalies, and neuromuscular disorders. Demographic characteristics included sex, age, BMI percentile, median income. The obstructive apnea hypopnea index (OAHI) was obtained from the PSG data. Moderate-Severe OSA was defined as OAHI³5 events/hour. Statistical analyses included chi-square tests, two sample t tests and non-parametric tests. Logistic regression was used for analysis of moderate-severe OSA and relevant predictor variables (age, sex, income and race/ethnicity)

Results

1745 adolescents with a mean age of 14.1±2.1 years were included, 56.1% were males and 43.9% were female. 58.2% Hispanic, 24.1% NH-Whites, 4.3% NH-Asians/Pacific Islander (Asian/PI), 4.2% NH-Blacks/African American (Black/AA) and 6.6% NH-Other. Male sex adolescents had higher OAHI (median 4.2 [IQR, 1.6-13.7] vs median 1.9 [IQR, 0.7-5.6], higher moderate to severe OSA than females (44.9% vs 27.2%, p-value< 0.05) and higher BMI percentiles (median 97.5 [IQR, 82.1-99.2] vs median 95.1 [IQR, 73.9-98.5], p-value< 0.01). Univariate logistic regression identified higher odds of moderate-severe OSA in Males (OR: 2.18, 95% CI: 1.78-2.66) compared to their female peers. This association persisted (OR: 2.24, 95% CI: 1.81-2.76) even after simultaneous adjustment for age, BMI percentile and median income. Finally, when stratifying by sex, we identified that Hispanic ethnicity (OR: 1.76, 95% CI: 1.19, 2.60) and NH-Asian race (OR: 2.36, 95% CI: 1.21, 4.59) were associated with increased odds of moderate-severe OSA only among males but not females, when compared to their NH-White peers.

Conclusion

In a sleep laboratory population, we identified higher moderate-severe OSA prevalence in male adolescents compared to females even when accounting for BMI and other relevant covariates. In addition, we found novel ethnic/racial disparities within the male sex population.

Support (if any)

NIMHD 1P50MD017344 and OT2 HL161847.
0774 Refining a Primary Care Navigation Intervention to Address Sleep Disparities: Caregiver and Clinician Perspectives
Authors: Uwah Eberechukwu , Abraham-Montgomery Michele , Nag Diya , Tapia Ignacio , Guevara James , Fiks Alexander , Williamson Ariel
Published: April 2024

Introduction

There are well-known racial, ethnic, and socioeconomic disparities in the incidence and treatment of pediatric sleep-disordered breathing (SDB). Addressing these disparities through culturally-tailored interventions could increase effectiveness and acceptability. As care navigation models have been beneficial in addressing modifiable patient and system-level barriers to care, we qualitatively explored participants’ perspectives of a sleep navigation (SN) program in primary care to inform future intervention refinement.

Methods

Thirty-three English-speaking caregiver-child dyads (child Mage= 7.1◻4.2 years; 51.6% girls; 100% Black, 3% Hispanic/Latinx; 93.5% maternal caregiver; 61.2% living ≤125% US poverty level) referred to SDB specialty care by their primary care provider were randomly assigned to clinical decision support (CDS) or to CDS with SN. SN was a 1-3 session program designed to support families in completing their SDB referral through motivational interviewing and care coordination strategies. Primary care clinicians (n=21) also participated in the study. We conducted semi-structured qualitative interviews with 10 caregivers (5 from CDS only, 5 from CDS + SN) and 9 clinicians to assess acceptability and perspectives on SN optimization. Interview data were coded and analyzed using thematic analysis.

Results

Overall, clinicians and caregivers that received SN reported strong acceptability. Participants also noted the benefits of racial concordance for families and navigators in transferring health knowledge to improve health literacy. Caregivers reported positive changes in their child’s sleep health and a better understanding of SDB. Themes related to SN refinement included adding tailored patient education such as audio-visual and written materials and enhancing strategic family-provider communication. Clinicians emphasized that SN could be enhanced beyond referral completion by including support for families needing additional SDB care (e.g., surgery) and through in-person, warm handoffs in clinics to build trust between families and navigators. Clinicians also recommended exploration of a broader navigation program that could integrate multiple primary care referrals and resource needs (e.g., food insecurity).

Conclusion

SN was perceived as highly acceptable by primary care clinicians and families of primarily Black and/or lower-income backgrounds. Sustainability of the resultant model requires additional refinement to determine optimal implementation strategies that integrate sleep navigation with other primary care needs.

Support (if any)

Children’s Hospital of Philadelphia Chair’s Initiative Round 8 (AAW).
0776 Polysomnography in Neonates with Myelomeningocele: Variability in Scoring and Interpretation
Authors: Mason Thornton , Hassan Fauziya , Shellhaas Renee , McCaffery Harlan , Lloyd Robin , Barks John , Chervin Ronald
Published: April 2024

Introduction

A prospective, longitudinal, multicenter study was designed to test the hypothesis that sleep-disordered breathing (SDB) is ubiquitous among newborns with myelomeningocele (MMC). We evaluated variability in polysomnography (PSG) scoring and interpretation.

Methods

Nine US pediatric centers participated. Neonates with MMC born at >30 weeks gestation underwent bedside PSG at ≥35 weeks postmenstrual age, before hospital discharge. PSGs were scored by registered technologists (RPSGTs) at each site using AASM Scoring Manual infant criteria and interpreted by a board-certified sleep medicine physician (BCSMP). A single, central RPSGT blinded to clinical outcomes then re-scored all PSGs. One of two pediatric BCSMPs reviewed each study; the two BCSMPs jointly determined diagnoses for consensus central reports. Differences in scoring and overall PSG interpretation were evaluated with kappa and intraclass correlation coefficients (ICC).

Results

For 110 neonates with MMC, the median apnea-hypopnea index (AHI) was 26.5 [11.3-43.3] as reported by sites, and 23.0 [IQR 13.0-44.8] as reported centrally (к 0.81). The central apnea index (CAI) was also scored similarly by sites (3.3 [0.9-7.0]) and centrally (2.6 [0.6-7.3]; к 0.88), as were hypopneas (15.2 [7.5;26.9]) and 14.1 [6.4;27.9]; к 0.82). However, the obstructive apnea index (OAI) was reported to be much higher by sites (10.0 [1.9-29.1]) than centrally (1.5 [0.5 – 4.1]; к 0.29). Moreover, overall PSG interpretation differed between sites and central reviewers. Obstructive sleep apnea (OSA) was reported as a diagnosis by sites for 57.6% of neonates, vs. 10.9% by central review (ICC 0.06); central sleep apnea was reported by sites for 26.6% of neonates, vs. 14.5% by central review (ICC 0.50). When hypopneas were the most prevalent respiratory events, central review favored a less specific diagnosis of sleep-disordered breathing.

Conclusion

Considerable variability in scoring of obstructive apneas, and in polysomnography-based diagnoses, may exist for neonates. Sites may have designated hypopneas as obstructive, driving the diagnosis of OSA, while central review noted equivocal features of neonatal hypopneas, leading to a less specific diagnosis of sleep-disordered breathing. Further work to develop infant respiratory scoring rules, and diagnostic criteria, and to evaluate their clinical use may be warranted.

Support (if any)

This study is supported by NIH (R01 HL147261).
0777 Respiratory Sleep Phenotypes in Prader-Willi Syndrome: Does the Type of Mutation Matter?
Authors: Queiroga Thereza , Lopes Maria-Cecilia , Spruyt Karen , Passone Caroline , Bueno Clarissa , Franco Ruth , Damiani Durval , Soster Leticia
Published: April 2024

Introduction

Prader-Willi Syndrome (PWS) is a congenital disorder with three types of mutation: deletion (DEL), maternal uniparental disomy (UPD), and imprinting center defects (IMP). Sleep breathing disorders and sleep disruption may change the clinical evolution of these patients. The study aimed to evaluate the clinical characteristics of PWS patients according to genetic mutation, characterizing their sleep phenotypes and focusing on sleep respiratory measures.

Methods

From a cohort (January 2014 to July 2021) 55 patients of the same research university center with confirmed PWS genetic diagnosis consented. We applied questionnaires for sleep disorders, physical examination, nasolaryngoscopy, and overnight polysomnography

Results

Participants were aged between 2 and 22 years with genetic confirmation for PWS: 28 DEL, 21 UPD, and 6 IMP, and 24 (43.6%) were male participants patients with UPD and DEL mutations underwent polysomnography. They were divided into three quartiles (Q1, Q2, and Q3) for each respiratory parameter: the apnea-hypopnea index (AHI), the time of SpO2 (minutes) during sleep (SpO2min), and T< 90%. The AHI values of Q3-DEL (AHI: 15.2) were higher than Q3-DUM (AHI: 10.3). The SpO2min in the DEL group was 62%, while in the UPD group it was 76%. Patients in the DEL group had a higher prevalence in longer quartiles with T< 90%.

Conclusion

PWS patients showed specific respiratory sleep phenotypes according to genetic diagnosis. We found that PWS patients with DEL had worse polysomnographic respiratory patterns. Further studies are needed to recognize the clinical implications of the sleep respiratory findings in the clinical endophenotype of PWS.

Support (if any)
0778 A Novel Endotype of Pediatric Obstructive Sleep Apnea Syndrome Revealed Using K-Means Clustering
Authors: Kim Seung Soo , Jang Han Na , Moon Jin-Hwa , Jeon Jin Yong , Ahn Dong Hyun
Published: April 2024

Introduction

With the recent emergence of precision medicine, endotype / phenotype research for sleep disorders has been increasing. However, there were still few related studies on pediatric obstructive sleep apnea syndrome (OSAS). The authors conducted this study to cluster pediatric OSAS patients using polysomnography (PSG) parameters and find new endotypes.

Methods

We retrospectively investigated the medical records of children and adolescents who had been diagnosed with OSAS (apnea-hypopnea index, AHI > 1/h on PSG) at the single sleep disorders center. We used age, total sleep time (TST), sleep latency (SL), REM sleep latency (RL), sleep efficiency (SE), wake time after sleep onset (WASO), and the percentage of stage N1/N2/N3/R (%N1/%N2/%N3/%R) for clustering. Prior to clustering, the dimensions of variables were reduced through uniform manifold approximation projection (UMAP). And then K-means clustering (KM) were used for clustering. We used average silhouette index (ASI) as evaluation metrics. After clustering, the differences among clusters were compared using one-way ANOVA.

Results

Total 306 subjects were enrolled (mean age 8.2 ± 4.1 years old, boy : girl = 194:112). There were statistical significant differences in pediatric daytime sleepiness scale (PDSS), children's depression inventory (CDI), and age (p-value < 0.01, respectively) among clusters. Sleep architectures, such as TST, SL, RL, WASO, and %N1/%N2/%N3/%R also showed a significance difference among clusters. There was no statistical significance in total AHI, SE, and sex. KM showed ASI 0.42 and revealed 4 clusters. Cluster 3 (daytime sleepiness with depressive mood, n=72) showed the higher PDSS (mean 13.9 ± 7.2) and CDI (mean 12.0 ± 9.6) than other clusters. Cluster 3 also showed the older age (mean 12.3 ± 3.1) than other clusters.

Conclusion

The authors revealed the novel endotype of pediatric OSAS accompanied by excessive daytime sleepiness and depressive mood regardless of AHI and SE, which were previously used as indicators of sleep quality. Further research is needed to identify new endotypes of pediatric OSAS that cannot be identified by the classic indicators.

Support (if any)

A grant of the Korea Health Technology R&D Project through the Korea Health Industry Development Institute (KHIDI), funded by the Ministry of Health &Welfare, Republic of Korea (grant number : RS-2023-00267049)
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

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 < 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

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 >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.

Support (if any)
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

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

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

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

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

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

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 (< 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.

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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

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<.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<.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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0793 Sleep Disordered Breathing in Children with Craniofacial Syndromes
Authors: Sivaram Ganeshraj , Jawad Kahir , El-Kersh Karim , Chariker Mark , Betz Kelly , Senthilvel Egambaram
Published: April 2024

Introduction

Children with Craniofacial Syndrome (CFS) are known to have a higher risk for Obstructive Sleep Apnea (OSA) due to various craniofacial abnormalities. We aim to assess OSA prevalence in a single cohort of children suffering from CFS and to stratify them by degree of OSA (mild, moderate, severe) and the overall management of OSA in CFS.

Methods

This was a single-center retrospective study that included children with confirmed CFS who underwent overnight polysomnography (PSG). OSA severity was categorized by obstructive apnea-hypopnea index (OAHI) as normal (< 1 event/hr), mild (1–4.9 events/h), moderate (5–9.9 events/h), and severe (≥ 10 events/h).

Results

PSG data were available for 46 children; 58.7% were male, with a mean age of 4.8 years. The average BMI was 18.2 (SD + 5.4 kg/m2) with an average z-score of 1.1 (SD + 4.0), and 76.1% of patients were Caucasian. Common referring symptoms were snoring (38.1%), witnessed apneas (29.1%), gasping/choking (17.4%), and oxygen desaturation (15.1%). The most common CFS was Pierre Robin Sequence (PRS) in 34.8%. OSA prevalence was 68.2% (23.3% mild, 20% moderate, 56.7% severe). The mean polysomnographic variables were total sleep time 346.2 min (± 120.0), AHI 22.2 (± 32.4), SaO2 nadir 83.8% (± 12.1), maximum CO2 level 54 mmHg (± 9.9), and arousal index 22.2 (± 23.4). The overall treatments consisted of mandibular distraction in 26.5%, continuous positive airway pressure therapy in 12.2%, tracheostomy in 6.1%, oxygen supplement in 2%, and lip-tongue adhesion in 2%.

Conclusion

In our cohort of children with CFS had a high prevalence of OSA with severe degree as the prevalent severity. PRS was the most common CFS and mandibular distraction was the most commonly performed procedure in infants with PRS.

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0794 Normative Transcutaneous Carbon Dioxide (TCCO2) Values in Older Children
Authors: Kalra Maninder , Deutsch Gabrielle , Karhoff Iris , Malerba Paola
Published: April 2024

Introduction

Transcutaneous carbon dioxide monitoring (TCCO2) is being increasingly used in pediatric sleep studies to assess for hypoventilation. However, consensus is lacking on a pediatric definition for hypoventilation. This study evaluates the applicability of adult hypoventilation definitions to healthy older children.

Methods

Subjects, 10 years to 18 years of age, participating in a study on sleep EEG dynamics in typical developing children were included. All participants underwent an in-lab nocturnal polysomnography with Natus Sleepworks TM system and following American Academy of Sleep Medicine (AASM) guidelines. Sentec monitor (Therwil, Switzerland) was used for TCCO2 recording. Studies were scored according to the AASM pediatric criteria. Nocturnal hypoventilation was evaluated according to the following definitions: – Over 25 percent of sleep time with a TCCO2>50 mmHg [AASM], – A peak of TCCO2>50 mmHg [S Ward, 2005], – A percentage of nighttime with a TCCO2>50 mmHg>2 % [R Paiva, 2009], – A TCCO2>10 mmHg above the morning awake minimal TCCO2 level [FJ O’Donoghue, 2003].

Results

Preliminary results on 7 subjects of which 71 % were females are presented. The mean age was 14.7 years (SD 2.6 years) and mean BMI was 22.2kg/m2 (SD 2.3). All subjects had an obstructive AHI of < 1; Mean awake TCCO2 was less than 45; Mean asleep TCCO2 was less than 50. There were no subjects with TCCO2 values >50 for over 25 % time or with delta TCO2 sleep to awake of >10 mmHg. There was one subject with peak TCCO2 values>50 and one subject withTCCO2 values >50 for 15 % time.

Conclusion

Our results, with no healthy child having >25 % of sleep time with TCCO2 over 50 mmHg, support the use of AASM hypoventilation definition in older children. When validity of AASH hypoventilation criteria is questionable due to sleep time < 4 hours or low REM sleep fraction, then an increase of sleep CO2 of 10 units as compared to awake values may also be used to identify hypoventilation.

Support (if any)

Research Institute, Nationwide Children's Hospital
0796 Implementation of the San Diego Sleep Survey (SDSS) in Children with down Syndrome
Authors: Bhattacharjee Rakesh , Wu F , Warner M , Inkelis S , Mung K , Stauffer Jacy
Published: April 2024

Introduction

Obstructive sleep apnea (OSA) is highly prevalent in children with Down syndrome (CWDS). Polysomnography is advised in all CWDS under four years, but at all ages, OSA prevalence remains as high as 75%. Polysomnography in CWDS requires skilled pediatric sleep labs, which are scarce. Implementation of comprehensive sleep questionnaires to identify sleep disorders could lead to earlier recognition and treatment however validated sleep questionnaires in CWDS are not widely available.

Methods

Caregivers of patients aged 1 to 21 years referred to Rady Children’s Hospital Sleep Center, San Diego, CA, completed the San Diego Sleep Survey (SDSS) via the Epic® EMR system. CWDS were identified using diagnostic codes (ICD-9/10). Additionally, surveys were collected from children without sleep complaints (control) at San Ysidro Children’s Dental Center and Chula Vista Medical Plaza. The SDSS, a 51-item scale, employed a 4-item Likert-type scale (Never, Sometimes, Usually, Do Not Know) for finely graded insights into sleep problems. Five domain scores assessed pediatric sleep problems: insomnia, sleep-disordered breathing (SDB), sleep disorder (e.g., parasomnia), sleep hygiene, and daytime symptoms (DS). A subset also completed the Pediatric Sleep Questionnaire (PSQ) and Children’s Habit Sleep Questionnaire (CSHQ) for comparative purposes.

Results

2103 patients completed SDSS (age 8.6±4.8) at the Sleep Center. Of these, 134 CWDS were identified (age 8.1±5.1; 58 females [43%]). Compared to controls (n=135, age 9.4±2.4), CWDS had significantly better hygiene scores (8.8±2.0 vs 9.9±2.3, p< 0.01), and significantly higher insomnia scores (14.4±3.8 vs 12.9±3.3, p< 0.01), SDB scores (17.3±5.2 vs 13.8±6.1, p< 0.01) and DS scores (23.6±6.7 vs 19.5±5.9, p< 0.01). Sixty-five CWDS completed the PSQ, which identified 27 children (43%) with a positive SDB score (PSQ Total Score ≥8), compared to 104 children (63%) with a positive SDB score on SDSS. Thirty-eight CWDS completed the CSHQ; all had a positive score (CSHQ Total Score ≥41).

Conclusion

The SDSS is a feasible method for evaluating sleep disturbance in CWDS, recognizing abnormalities in various domains including SDB. Future analyses will compare SDSS to PSQ and CSHQ to assess convergent validity for screening for sleep disorders. Sensitivity and specificity of SDSS to identify OSA compared to polysomnography will be explored.

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0797 Afraid to Sleep Alone: Characteristics of Parents and School-age Children Who Cannot Sleep Independently
Authors: O'Grady David
Published: April 2024

Introduction

A surprising number of school-age children (5 - 12) do not sleep independently. Every night, the child sleeps in the parent's bed, or a parent sleeps in the child’s bed. This habit eventually becomes very frustrating for parents. But when they try to get their child to sleep alone, the child becomes intensely anxious and vigorously resists. The ensuing struggle can bring chaos to family routines and distress to everyone. The problem is surprisingly common, occurring in up to 10% of school-age children. Nevertheless, it has received little systematic attention.

Methods

1,630 parents completed an online survey. All the parents had a school-age child (5 to 14 years) who routinely co-slept with a parent (in the parents’ bed or the child’s bed). The parents identified the habit as a problem. Parents accessed the survey alongside an accompanying online article about this sleep habit.

Results

The overwhelming majority of children (>67%) had never learned to sleep alone. Most parents reported that the problem was very distressing to them, disrupted their sleep, disrupted family routines, and interfered with sexual intimacy. When parents tried to get their child to sleep alone, the child displayed extremely elevated anxiety and protest, even panic. Parents’ efforts were unsuccessful and left them frustrated and worried about their child’s psychological health. Yet 81% of these children had never received any mental health treatment. Among mothers, about 63% reported they had a history of anxiety disorder themselves, twice the rate of the general population. Among fathers, the rate of reported anxiety disorder was about the same as in the general population. These findings imply that clinical interventions with this population need to specifically address mothers' challenges with anxiety.

Conclusion

Challenges in transitioning from co-sleeping to sleeping alone occur commonly, disrupt routines, and cause marked distress. These dynamics can be understood as involving sleep associations, parental accommodation, ineffective limit setting, and anxiety escalation. Behavioral interventions can be very effective. This presentation will describe one such approach that typically resolves the problem in less than two weeks and can be implemented by parents acting on their own as they follow a treatment guideline.

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0798 Feasibility & Acceptability of SAFE: A Brief Trauma-Informed Behavioral Sleep Intervention for Youth in Foster Care
Authors: Rech Megan , Harmouch Sirine , Myers Annika , Kim Jinu , Cifre Anthony , Vieira Alyssa , Andre Theo , Alfano Candice
Published: April 2024

Introduction

Children who have spent time in foster care unsurprisingly experience high rates of sleep problems and disorders, which negatively impacts well-being. Sleep and Adjustment in Foster Environments (SAFE) is a brief, telehealth-delivered behavioral sleep intervention for caregivers of young children in or adopted from foster/kinship care. Building upon existing evidence-based behavioral sleep strategies by using a trauma-informed approach, SAFE promotes children’s self-regulation, felt safety, and connection with foster caregivers to improve sleep. Using data from a randomized controlled pilot study, we explored the feasibility and acceptability of SAFE compared to an active control condition (Sleep Education Support; SES), which provided families with a mailed informational booklet about young children’s healthy sleep.

Methods

Foster/kinship caregivers and adoptive foster parents of children ages 2-5 years were recruited across Texas. Children with organic sleep disorders (e.g., OSA), major medical conditions/disorders, and/or autism were ineligible. Beliefs about treatment were assessed pre-treatment with the Credibility/Expectancy Questionnaire; perceived intervention utility and acceptability were assessed post-treatment with the Client Satisfaction Questionnaire and via qualitative interviews.

Results

Over an 18-month period, N=127 families inquired about the study, 78 were screened, and 67 were eligible. Of these, 22/67 (33%) did not move forward due to scheduling difficulties (n=9), no longer being interested (n=8), or loss of contact (n=5). The remaining 45 eligible families were randomized to SAFE (n=22) or SES (n=23). Among SAFE families, 21/22 (95%) completed all three intervention sessions and the post-treatment assessment. Among SES families, 20/23 (87%) completed the post-intervention assessment. Primary factors influencing study withdrawal included the child leaving the home and scheduling difficulties. Results of independent-samples t-tests revealed significantly higher treatment satisfaction in the SAFE group (M=29.57, SD=4.78) compared to the SES group (M=23.95, SD=7.00; t=-2.988, p=.005). Groups did not significantly differ in treatment credibility or expectancy.

Conclusion

SAFE appears feasible and acceptable to foster/adoptive caregivers. While both groups reported similar baseline beliefs about treatment and shared positive post-treatment feedback, satisfaction was greater for the SAFE group. Results underscore a need for sleep health interventions tailored to this population.

Support (if any)

This study was funded by the Grant to Enhance and Advance Research (GEAR) from the University of Houston.