bluuebunny/crossref_metadata_2025 huggingface dataset:
494 Placebo-Controlled, Double-Blind, Randomized Withdrawal Study of Lower-Sodium Oxybate in Adults With Idiopathic Hypersomnia
Authors: Dauvilliers Yves , Arnulf Isabelle , Foldvary-Schaefer Nancy , Chandler Patricia , Parvataneni Rupa , Chen Dan , Skobieranda Franck , Bogan Richard
Published: May 2021

Introduction

Idiopathic hypersomnia (IH) is a rare central hypersomnolence disorder characterized by excessive daytime sleepiness, prolonged nighttime sleep, and sleep inertia. No US/EU medication is approved for treatment of IH. Lower-sodium oxybate (LXB; Xywav™; previously designated JZP-258) is a novel oxybate treatment with 92% less sodium than sodium oxybate (Xyrem®). The efficacy and safety of LXB was evaluated in adults with IH.

Methods

Eligible participants aged 18–75 years with IH began once- or twice-nightly LXB treatment entering an open-label titration and optimization period (10–14 weeks), followed by a 2-week, open-label, stable-dose period (SDP); they were then randomized to placebo or to continue LXB treatment during a 2-week, double-blind, randomized withdrawal period (DBRWP). The primary efficacy endpoint was change in Epworth Sleepiness Scale (ESS) score; key secondary endpoints were proportion of participants who reported worsening (minimally/much/very much worse) on Patient Global Impression of Change (PGIc) and change in Idiopathic Hypersomnia Severity Scale (IHSS) score, all from end of SDP to end of DBRWP.

Results

The study enrolled 154 participants (mean±SD age, 40±14 years; 68% female; mean±SD ESS, 16±3.6); mean±SD dose was 6.0±1.6 g/night. Mean±SD ESS score (n=115) decreased over open-label titration/optimization (15.7±3.8 at baseline, 9.8±4.5 at week 4, and 6.1±4.0 at the end of the SDP). At the end of the DBRWP, significant worsening was observed in participants randomized to placebo, compared with maintenance of improvement in participants randomized to continue LXB, in ESS scores (n=115; LS mean difference [95% CI] in change from SDP, −6.51 [−7.99, −5.03]; P<0.0001), in the PGIc (88.1% for placebo vs 21.4% for LXB; P<0.0001), and in IHSS scores (estimated median difference [95% CI], −12.00 [−15.0, −8.0]; P<0.0001). Common adverse events (AEs) included nausea (21.4%), headache (16.2%), anxiety (14.9%), dizziness (11.7%), insomnia (11.7%), and vomiting (10.4%). Serious AEs occurred in 4 participants (non-cardiac chest pain, rhabdomyolysis, syncope, and nephrolithiasis/pyelonephritis); none were reported related to study drug.

Conclusion

In participants with IH, LXB demonstrated a clinically meaningful effect on excessive daytime sleepiness, self-reported global change, and overall IH symptom severity. The overall safety profile was consistent with that of LXB in narcolepsy.

Support (if any)

Jazz Pharmaceuticals
498 It Makes Relationships Harder: The Role of Narcolepsy in Social and Romantic Relationships in Young Adults
Authors: Davidson Ryan , Biddle Kelsey , Scammell Thomas , Nassan Malik , Zhou Eric
Published: May 2021

Introduction

Narcolepsy typically begins during adolescence/young adulthood – a crucial period for developing relationships. As symptoms would be expected to impact social interactions, we studied how narcolepsy impacts social, romantic, and sexual relationships in young adults.

Methods

Young adults (18–39 years; N=262) with a self-confirmed narcolepsy diagnosis were recruited through national narcolepsy patient organizations. Participants completed an online survey, including open-ended questions, to evaluate their social, romantic, and sexual relationships and communication with medical providers.

Results

Participants (mean=28.5 years old; SD=5.6) were primarily female (87%), White/Caucasian (92%), employed (75%), and had Narcolepsy Type I (56%). All participants indicated that narcolepsy made social life or entering relationships somewhat or a lot more difficult. Rates of cohabitation and marriage were comparable to nationally representative samples. On the Couples Satisfaction Index, most participants reported feeling satisfied with their romantic relationships (mean=15.8; SD=4.3). On the Multidimensional Scale of Perceived Social Support, participants reported receiving greater support from their significant others (mean=4.5; SD=1.4) compared to both family (mean=3.7; SD=1.5; p<.05) and friends (mean=3.9; SD=1.4; p<.05). There was no difference between family and friends (p>.05). Eighty-percent of participants indicated that narcolepsy impacted their sex life, including experiencing cataplexy or falling asleep during sex. Few participants indicated that their providers asked about their social (31%) and sex life (10%). In contrast, 73% of participants wanted providers to ask about social life and 45% wanted providers to ask about sex life.

Conclusion

Narcolepsy substantially impacts social functioning in young adults. They often prioritize the development of a single, meaningful romantic relationship as their disorder makes sustaining more social relationships challenging (“I do not go out and socialize at all, but spend all my time at home nurturing my romantic relationship because it’s the most important to me”). At a time of sexual development, there are considerable implications of narcolepsy symptoms on their sexual experiences. Though many participants were interested in discussing their social, romantic, and sexual relationships with medical providers, only a small proportion of providers inquire. Effective treatment of narcolepsy in young adults should include support for the impact of the disorder on relationship health.

Support (if any)

Jazz Pharmaceuticals.
505 Efficacy of Pitolisant in the Treatment of Cataplexy in Adults With Narcolepsy
Authors: Davis Craig , Zarycranski Donna , Doliba Markiyan , Dayno Jeffrey , Schwartz Jean-Charles
Published: May 2021

Introduction

Pitolisant was initially approved by the FDA in 2019 for the treatment of excessive daytime sleepiness in adult patients with narcolepsy; in 2020, the indication was expanded to include the treatment of cataplexy.

Methods

Cataplexy data from 7- or 8-week, randomized, placebo-controlled studies (HARMONY-CTP, HARMONY-1) are reviewed and summarized. In HARMONY-CTP, all patients were required to have ≥3 cataplexy attacks per week at baseline; HARMONY-1 enrolled patients with narcolepsy with or without cataplexy. Pitolisant was individually titrated to a maximum potential dose of 35.6 mg/day. The weekly (WRC) or daily (DRC) rate of cataplexy attacks was calculated from patient diaries.

Results

In HARMONY-CTP (pitolisant, n=54; placebo, n=51), mean baseline WRC was 11.7 in the pitolisant group and 9.6 in the placebo group. In the subset of HARMONY-1 patients with cataplexy (pitolisant, n=17; placebo, n=11), mean baseline DRC was 1.5 and 1.2, respectively. In HARMONY-CTP, least-squares (LS) mean change in WRC was significantly greater for pitolisant versus placebo at Week 2 (-4.1 vs 1.2; P=0.004) and continued through end of treatment (Week 7; -6.5 vs -0.1; P<0.001). In HARMONY-CTP, treatment response was observed in 66.7% of pitolisant-treated versus 25.5% of placebo-treated patients (P<0.001) for WRC reduction ≥50%, and 77.8% versus 33.3% of patients (P<0.001) for WRC reduction ≥25%. In HARMONY-1, LS mean change in DRC was significantly greater for pitolisant versus placebo at Week 5 (-1.04 vs 0.17; P=0.047) and continued through end of treatment (Week 8; -0.96 vs 0.35; P=0.035). In a pooled analysis of patients with high burden of cataplexy (≥15 attacks/week) at baseline (pitolisant, n=20; placebo, n=11), LS mean change in WRC at end-of-treatment assessment was significantly greater for pitolisant (-14.5; baseline, 23.9; final, 9.4) versus placebo (-0.1; baseline, 23.1; final, 23.0; P=0.004). There was no evidence of rebound cataplexy after a 1-week placebo washout period.

Conclusion

Pitolisant, at once-daily doses of up to 35.6 mg, demonstrated a statistically significant and clinically meaningful reduction in the frequency of cataplexy attacks in adults with narcolepsy, including patients with a high symptom burden. Onset of response was observed within the first few weeks of pitolisant treatment.

Support (if any)

Bioprojet Pharma and Harmony Biosciences, LLC.
520 A Cone-Beam Computed Tomography Study on the Impact of Bruxism in Craniomandibular Morphology of Adults. Preliminary findings
Authors: Leite Irene , Rode Sigmar , Lopes Sérgio , Meira e Cruz Miguel , Oliveira Wagner
Published: May 2021

Introduction

Although bruxism, either during awake and during sleep is still a controversial topic in contemporary Medicine, its prevalence and its putative relationship with several conditions interfering with health and wellbeing make it relevant as a target to be studied in distinct scientific domains. Histological and morphological on the craniomandibular complex (CC) have been documented with different techniques. However, there is a lack of high quality imaging studies showing structural changes among the different anatomical structures therefore leading to a misinterpretation of the real impact of bruxism on such elements.The aim of this clinical retrospective comparative study was to investigate whether there are structural differences on the craniomandibular complex of bruxers in face to non-bruxers evaluated by a cone-beam computed tomohraphy (CBCT). We present the preliminary findings of this still ongoing study.

Methods

A retrospective analysis of CBCT imaging exams from patients with ages between 18 and 44 years old, were distributed among 1) bruxism group:BG (n=31); 2) Non-bruxism/Control group (n=21). Bruxism diagnosis followed non-instrumental approach suggested by the bruxism consensus 2013. To the craniomorphological evaluation, 3D CBCT SYM Diagnosis protocol was implemented by a calibrated expert at an adequate local with reduced light. Photograpic parameters were analyzed according to standard procedures.

Results

Female gender prevailed without differences between groups (61,3% in BG and 66,7 in CG; p=0,91). Age of BG was significantly higher than CG (40,1 yo versus 28,4;p=0,002). Mandibular structure as inferred by its shape was significantly higher in BG compared with CG.

Conclusion

In this preliminary study the CBCT 3D image showed important and significant changes in mandibular structure in bruxers compared with non-bruxers.

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527 Patient-Reported Treatment Outcomes in the Mayo Clinic REM Sleep Behavior Disorder Registry: Efficacy of Melatonin and Clonazepam
Authors: Grayson Kammi , Gossard Thomas , Teigen Luke , Feemster John , McCarter Stuart , Timm Paul , Sandness David , Carvalho Diego , Junna Mithri , Lipford Melissa , Olson Eric , Tippmann-Peikert Maja , Boeve Bradley , Silber Michael , St. Louis Erik
Published: May 2021

Introduction

REM sleep behavior disorder (RBD) is characterized by disruptive, violent dream enactment behaviors (DEB), necessitating symptomatic treatment to prevent injury and reduce DEB frequency and severity. Melatonin and clonazepam are regarded as RBD therapeutic mainstays, although outcomes data remains limited. We surveyed RBD patients to determine their outcomes following melatonin, clonazepam, and melatonin-clonazepam combination therapy.

Methods

Mayo Clinic RBD Patient Registry participants received an electronic survey concerning treatment type(s) and dose(s), efficacy, and adverse effects. The primary outcome was treatment efficacy, determined by comparing DEB frequency/severity ratings on a visual analog scale (VAS). Adverse effects severity was assessed by Likert scales. We comparatively analyzed VAS before and after treatment and adverse effects between treatments using non-parametric statistical tests.

Results

Sixty-eight of 109 patients responded (62.3%; 64 had analyzable data) with a mean age of 67.7 years. Fifty-seven (85%) were men, with mean RBD symptom duration of 13.9 years. Patients receiving each treatment were: melatonin=30, clonazepam=8, and combination=12; 14 received other or no treatment. Baseline VAS ratings were similar between groups. Only melatonin (p=0.003) and combination therapy (p=0.039) improved VAS ratings; clonazepam monotherapy did not improve VAS. Only melatonin monotherapy was reported to lower VAS compared to untreated patients (p=0.02). Optimally effective mean dosages were melatonin 9.95±5.06 mg and clonazepam 0.81±0.48 mg. Patient frequencies reporting one or more moderately-severe side effect(s) were similar between melatonin (15%), clonazepam (7%), and combination therapies (9%). Twenty-five (36.8%) patients had received only one medication trial, while 41.2% required more than one medication. Of these, 15 (22.1%) tried 2 and 13 (19.1%) tried 3 or more treatments.

Conclusion

Melatonin therapy at an approximate mean 10 mg dosage improved patient-reported DEB frequency/severity on VAS, compared between both previous intraindividual baseline ratings and with untreated patients, while clonazepam monotherapy did not, without differential adverse effects. Clonazepam monotherapy data were limited. These data inform future prospective melatonin symptomatic therapy trials for RBD. Additionally, 41.2% required more than one RBD pharmacological treatment, suggesting a current therapeutic gap and unmet need for future development of biologically-informed, evidence-based symptomatic RBD therapeutics.

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538 Sleep and Self-Efficacy: The Role of Domain Specificity in Predicting Sleep
Authors: Ghose Sarah , Dzierzewski Joseph , MacPherson Ashley , Sabet Sahar , Williams Claire , Reid Morgan , Dautovich Natalie
Published: May 2021

Introduction

Poor sleep occurs across the lifespan and has a number of consequences across biopsychosocial life domains. As such, it is important to identify modifiable psychobehavioral mechanisms contributing to sleep disturbance as potential targets for research and intervention. Self-efficacy may play a significant role in sleep behavior outcomes. The present study aimed to determine the differential associations between general and sleep self-efficacy and sleep among a lifespan sample of adults.

Methods

Data from the Investigating Sleep Longitudinally Across Normal Development (ISLAND) study were utilized for the present investigation. Participants were 3,284 adults (48.5% female, 6.4% other-identifying, Mage= 43 yrs., SD=16.72 yrs.) who completed self-report sleep, general-, and sleep self-efficacy measures as part of their study involvement. A Structural Equation Model (SEM) was estimated to investigate whether general or sleep self-efficacy were significant predictors of a latent sleep disturbance variable, which was comprised of the presence of insomnia symptoms (Insomnia Severity Index) and the absence of sleep health (RU-SATED). Invariance tests were utilized to determine whether the model held across age and sex.

Results

The structural model evidenced good fit to the data and indicated that general self-efficacy did not have a significant, direct impact on the latent sleep construct (b=-.01, p=.33); however, sleep self-efficacy did display a significant, negative association with sleep disturbance (b=-.82, p<.001), whereby increased sleep self-efficacy was associated lower levels of disturbed sleep. The model was largely invariant across age and sex.

Conclusion

Sleep self-efficacy surfaced as an important predictor of sleep disturbance above and beyond general self-efficacy. Findings highlight the importance of domain specificity in the predicting sleep outcomes. Additionally, findings suggest the need for increased research into and application of interventions targeted toward increasing sleep self-efficacy in individuals with sleep disturbance as a potential avenue to improve sleep health.

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549 Associations of Bedtime, Wake-time and Employment Status by Gender and Race
Authors: Lyu Xiru , Dunietz Galit Levi , O’Brien Louise , Chervin Ronald , Shedden Kerby
Published: May 2021

Introduction

Poor sleep quality has been reported in the unemployed compared with employed. How sleep varies by employment status has been rarely examined at a population level. Therefore, we investigated sleep-wake patterns among employed, unemployed but actively seeking a job, and not-in-the-labor-force participants by gender and race/ethnicity.

Methods

Methods We used data from the American Time Use Survey (ATUS), a nationally representative sample of US residents aged ≥15years, which records weekday/weekend activities in a 24-hour period (4:00am-4:00am). This sample was restricted to participants aged 25–60 years (n=130,062). This analysis utilized functional nonparametric regression based on dimension reduction and neighborhood matching. We modeled the relationship between participant-specific sleep-wake trajectories, coded by minute, and employment status. Implementing the counterfactual approach, we estimated the effects of each employment scenario on participant-level expected sleep trajectory. This approach allowed the examination of hypothetical sleep-wake trajectories for each participant if their employment status differed from the observed. We then marginalized these findings to gender and race/ethnic subpopulations, controlling for confounders and secular trends.

Results

Mean age was 42□0.01 years, nearly half (51%) of participants were women and 68% were Whites. The proportions of employed, unemployed, and not-in-the-labor-force were 79%, 16.5% and 4.5%, respectively. On average, unemployed and not-in-the-labor-force participants had a later bedtime and wake-time compared with employed. With the exception of Whites, each individual race/ethnicity group showed pronounced differences in sleep-wake patterns by employment status. Of note, the likelihood of still being asleep up to 9:00am was greater when unemployed in comparison to had they been employed. Compared with employed, differences in sleep-wake patterns were pronounced among Blacks and Hispanics had they been unemployed, but attenuated if they were out-of-the-labor-force. Gender alone was not a strong moderator of the relationship between sleep-wake patterns and employment status. Unemployed participants had bedtime after 11pm, regardless of gender or race/ethnicity.

Conclusion

Using the counterfactual approach, we predicted sleep-wake patterns among individuals had they been employed, unemployed, or out-of-the-labor-force by gender and race/ethnicity. Though cross-sectional, our data suggest that the sleep schedules of racial/ethnic minorities in comparison to Whites may be more affected by employment status.

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551 Outcomes of Sickle Cell Crisis in Relation to Pediatric Obstructive Sleep Apnea in the United States: A US Population cohort study
Authors: Patel Palakkumar , Munoz Tomas , Adavadkar Pranshu
Published: May 2021

Introduction

This study aimed to quantify the impact of obstructive sleep apnea (OSA) on the mortality, morbidity, and resources utilization among children admitted with Sickle cell crisis (SCC)

Methods

This is a retrospective analysis using the 2016 and 2017 National Inpatient Sample Database. The Inclusion Criteria was a principal Diagnosis of SSC and age <18 years. OSA, as a Secondary Diagnosis, was identified using the appropriate ICD-10 CM codes. The Primary Outcomes were Inpatient Mortality, and secondary outcomes were: In-Hospital Length of Stay(LOS), Total hospitalization Cost, Blood transfusion (BT) requirement, and a requirement for Invasive mechanical ventilation (IMV). We used Multivariate Linear/ logistic regression to adjust for confounders including age and sex.

Results

Out of 36,484 children with SSC included in the study, 1450 children had OSA (SCC+OSA). SSC-OSA and SSC+OSA groups did not differ in gender, household income, and hospital characteristics, but did so in age (11.3 vs 12.4; p <0.001). OSA was most common in the age group of 13–18 (54%) and lowest in 0–4 (2.4%). Compared to SSC-OSA, the SCC+OSA cohort had significantly higher odds of mortality (adjusted OR= 11.9, [95% Confidence Interval: 1.02- 138.8],p=0.04). Additionally, SSC+OSA cohort was associated with increased odds of IMV (aOR=5.24 [CI: 1.84 – 14.8], p=0.002), longer LOS (adjusted mean difference (aMD)=0.67 [CI-0.32 – 1.02], p=<0.001), and higher hospitalization Cost (aMD=2818.76 [CI-1680- 4157], p=<0.001). No difference in BT (aOR=0.94 [CI: 0.68 – 1.29], p=0.71) was noted.

Conclusion

This study demonstrates that the presence of OSA is associated with detrimental outcomes in SSC with higher in-hospital mortality, higher morbidity (Invasive mechanical ventilation rate), and higher resource utilization (LOS, total hospitalization cost). More attention to the screening, early diagnosis, and appropriate treatment of OSA is imperative to improve health outcomes in children with sickle cell disease.

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553 Assessment of sleep disorders in children and adolescents with obesity
Authors: Zarpellon Raquel Simone Maccarini , Vilela Regina Maria , Radominski Rosana Bento , de Souza Crippa Ana Chrystina
Published: May 2021

Introduction

When studying the inherent aspects of sleep it is important to assess how the quality and quantity of sleep in the last two decades may be one of the reasons for the increase in childhood obesity, which has been growing rapidly worldwide. This study aims to assess the presence of sleep disorders in overweight children and adolescents.

Methods

An descriptive study was conducted with data collection from 43 patients between 6 and 13 years old diagnosed as overweight. They were patients of a specialized service for children and adolescents with obesity that is part of the Hospital de Clínicas of the Federal University of Paraná, located in Curitiba, Brazil. To investigate the presence of sleep disorders, the Sleep Disturbance Scale for Children (SDSC) questionnaire was administered. The factors assessed were: Disorders of Initiating and Maintaining Sleep, Sleep Breathing Disorders, Disorders of Arousallnightmures, Sleep Wake Transition Disorders, Disorders of Excessive Somnolence and Sleep Hyperhydrosis.

Results

The mean age of the patients that took part in the research was 10 years and 7 months (± 1.95). The mean BMI of the participants was 29.57 kg/m2 (± 4.38), the majority being diagnosed with obesity. The sum of all SDSC factors demonstrated the presence of pathological sleep in 58.1% (25) of the sample, whereas 51.2% (22) of the patients had Sleep Breathing Disorders and 58.1% (25) had the Sleep Wake Transition Disorder.

Conclusion

The present study demonstrated the presence of sleep disorders in overweight children and adolescents. As for Sleep Respiratory Disorder, a situation has already been advocated in the current literature for this audience. In relation to the Sleep-Wake Transition Disorder and pathological sleep, further research is needed to prove the presence of the disorder in other groups studied. Here is the suggestion that future research be done with subjective and objective data collection on sleep within a larger sample, in order to confirm the association between sleep disorders and childhood obesity.

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554 Utilization of Pediatric Sleep Questionnaire to Screen for Obstructive Sleep Apnea in a Difficult-to-treat Asthma Cohort
Authors: Lin Nancy , Hantragool Sumalee , Huang Guixia , Hossain Md Monir , McDowell Karen , Guilbert Theresa , Kercsmar Carolyn , Simakajornboon Narong
Published: May 2021

Introduction

Early detection and management of obstructive sleep apnea(OSA) could improve asthma control in children with difficult-to-treat(DTT) asthma. The purpose of this study was to assess the effectiveness of the pediatric sleep questionnaire(PSQ) to screen for OSA in children with DTT asthma, and to compare clinical characteristics between those with positive and negative PSQ scores.

Methods

A prospective study of 81 children with DTT asthma was completed from 2015–2017. The PSQ, Epworth sleepiness scale(ESS), Pediatric Quality of Life(PQL), and the academic performance questionnaires(APQ) were administered during clinic visits. Polysomnography(PSG) was recommended for PSQ score>0.33. Medical records were reviewed for asthma clinical characteristics. The cohort was separated into positive(PSQ>0.33) and negative PSQ score(PSQ≤0.33) groups for analysis.

Results

The mean age of the cohort was 11.3±4.5 years and the mean body mass index was 22±7.6 kg/m2. Sixty-two percent were male and 68% were African-American. Forty-nine(53%) subjects had positive PSQ (0.5[0.4 – 0.7]). The positive group had higher ESS score (10.5[8 – 13] vs. 6[2 – 8], p<0.0001) and lower total PQL score (58.7[47.8–72.8] vs. 79.4[70.7 – 87], p<0.0001) than the negative group. There was no difference between APQ scores(p=0.07). The positive group had lower asthma control test(ACT) scores than the negative group (17.5[15 – 20.5] vs. 21[19 – 22], p<0.0001). Furthermore, the positive group was more likely to have gastroesophageal reflux (OR: 3.97, 95%CI: 1.7 to 9.1, p=0.0018). Twenty-nine(59%) subjects in the positive group had subsequent PSG, and 17(58.6%) subjects were diagnosed with OSA (14 mild OSA, 1 moderate OSA, 2 severe OSA). The mean obstructive index in the positive group was 3±5.5 events/hour. There was 1 subject with central apnea and alveolar hypoventilation. Of the 17 subjects with OSA, all received treatment with nasal steroids, 3 were treated with non-invasive positive pressure ventilation, and 4 had surgical intervention.

Conclusion

Children with DTT asthma who have positive PSQ have higher degree of daytime sleepiness, lower quality of life and worse asthma control. The positive group was more likely to have GERD, which may suggest a relationship between nighttime asthma symptoms and OSA. Further studies are needed to evaluate the effects of OSA treatment on asthma control.

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560 Infant OSA: Impact on Growth and Development Outcomes
Authors: Kochhar Rashi , Pereira Nicola , Liu Katie , Morse Elliot , Veler Haviva , Maresh Alison
Published: May 2021

Introduction

Obstructive sleep apnea (OSA) related growth and developmental deficits were previously described in young children. However, growth and development outcomes of OSA in infants is not as clear.

Methods

We retrospectively reviewed polysomnography (PSGs) of infants 0–12 months of age done at one tertiary care, urban medical center, between 2012–2019. Indications for the studies were symptoms or high risk for OSA. Diagnosis of OSA was determined when obstructive apnea hypopnea index (oAHI) >1/hr. Demographic information and physician diagnosed developmental and growth delays were collected from medical records.

Results

A total of 83 infants were included, mean age was 4.4months (6 days-12 months). Mean baseline oAHI was 16.8/hr (1–91.46/hr). 23 patients were noted to have failure to thrive (27.7%), 27 patients had general developmental delay (32.5%), 22 patients had speech delay (26.5%), and 9 patients had motor delay (10.8%). Out of all the patients with any form of delay (N=46), 39.13% were premature, 71.73% had an underlying neurologic abnormality and 39.13% had some other underlying diagnosed syndrome. 2 patients had delay but no associated comorbidity. Patients without any developmental delay showed trend towards likelihood of more severe OSA compared to patients with any delay (p=0.0455).

Conclusion

Infant OSA is a separate entity from pediatric OSA and requires a better understanding of its association with developmental outcomes. Infants are particularly vulnerable to obstructive sleep-disordered breathing related to their upper airway anatomy, adverse pulmonary mechanics and a REM-predominant sleep state distribution. We describe a cohort of 83 infants undergoing PSGs in their 1st year of life with developmental and growth follow up until 2nd year of life. Most of the infants with moderate to severe OSA had some delay in their development. Infants without diagnosed delay, were less likely to have severe OSA. It remains unclear to what degree OSA is responsible for these findings as opposed to pre-existing comorbidities. Additional prospective, controlled studies with standardized developmental assessments are warranted to assess causality.

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568 Sleep Evaluation in Down Syndrome: What is the Adherence to American Academy of Pediatrics DS Guidelines?
Authors: Morse Anne Marie
Published: May 2021

Introduction

Specialized health care guidelines for children with Down Syndrome (DS) published by the American Academy of Pediatrics (AAP) provided specific recommendations based on the higher risk needs of individuals with DS. Obstructive sleep apnea (OSA) is reported to be present in 50–79% of individuals with DS. According to the AAP guideline, all individuals with DS should have a polysomnography (PSG) evaluating for OSA by 4 years old and then screened by history and physical exam annually thereafter. An interim analysis of an ongoing Down Syndrome Research study was evaluated to determine rate of adherence to these guidelines.

Methods

The Dimensional, Sleep, and Genomic Analyses of Down Syndrome to Elucidate Phenotypic Variability study enrolled down syndrome patients 30 months and older, as well as first degree relatives to participate. Patients completed a standardized clinical sleep interview, childhood sleep habits questionnaire and was asked to complete 2 week sleep diary, actigraphy and polysomnography. We aimed to characterize the rate of PSG completion by 4 years of age, number of research PSGs completed and rate of OSA identified on research PSG.

Results

A total of 31 patients were consented. The median patient age was 10 years old with a slight female predominance (15F:12M). 27 patients completed the sleep interview and 19 successfully completed a scorable polysomnography. Only 7 patients had completed a PSG previously by age of 4 years. 11 of 19 studies demonstrated obstructive sleep apnea ranging from mild to severe severity (1.7–42.5/hr). REM AHI (range 1.2–58.2/hr, mean 19/hr and median 12.3/hr) demonstrated increased severity.

Conclusion

Despite AAP guidelines recommending universal PSG evaluation by the age of 4 years of age, only 26% of patients interviewed has a PSG successfully completed previously. Additional recommendations by AAP include yearly surveillance of symptoms although there is poor correlation between parent report and polysomnogram results. Of the 19 research completed PSGs, 58% demonstrated OSA with the mean and median results consistent with moderate to severe OSA and worsening during REM sleep. Improved effort to successfully obtain PSG in this population is needed. Further study is ongoing to evaluate the relationship to other health and cognitive outcomes.

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NIMH
570 Comparing NIV Adherence in Early versus Advanced Stage Sleep Disordered Breathing for Children with Neuromuscular Disease
Authors: Bedi Prabhjot , Hasbun Guillermo , Castro-Codesal Maria
Published: May 2021

Introduction

Children with neuromuscular disease (NMD) typically develop progressive sleep disordered breathing (SDB), including obstructive sleep apnea (OSA), nocturnal hypoxemia and/or hypoventilation, due to loss of upper airway muscle tone and weakness of respiratory muscles. Commonly, the SDB initially presents during rapid eye movement (REM) sleep, as this stage is associated with physiological muscle atonia, but then progresses to non-REM (NREM) sleep and ultimately daytime respiratory insufficiency. Non-invasive ventilation (NIV) is currently the treatment of choice for children with NMD and SDB. However, the use of NIV in REM-related SDB is less demonstrated and adequate therapy adherence is unclear. The aim of this study is to determine differences in NIV adherence in children with early (REM) versus advanced (non-REM) SDB.

Methods

Children (0–18 years) diagnosed with NMD and using NIV for the past 10 years were included. Demographic, clinical, technology-related, and sleep study data were collected from medical charts and polysomnography reports. Adherence data (mean hours of NIV use and % days NIV was used >4hrs) were collected from NIV machine downloads. Children were categorized into two groups based on based on their apnea-hypopnea index (AHI) ratio between REM and NREM sleep. Children with REM-SDB were defined as a REM/NREM AHI ratio of ≥ 2. Children with NREM-SDB were defined as a REM/NREM AHI ratio < 2.

Results

A total of 14 children (9 REM-SDB and 5 NREM-SDB) were included in the analysis. Both groups were comparable with respect to demographic, clinical, and technology-related characteristics. A total of 24 adherence reports were available for the cohort (16 REM-SDB and 8 NREM-SDB). The mean hours of NIV use per night was comparable between the REM-SDB and NREM-SDB groups (9.2±1.3hrs vs. 9.0±0.4hrs respectively), but the percent days NIV was used >4hrs was higher in the NREM-SDB group (68.7±9.6 vs. 93.0±2.7, p=0.03).

Conclusion

NIV adherence was high for children with both REM-SDB and NREM-SDB. While hours of NIV use were comparable between both groups, suggesting good NIV tolerance through the night, children with REM-SDB had a lower percentage of days with NIV use >4hr, suggesting less willingness to use the therapy.

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572 Utility of Split Night Polysomnograms in Children
Authors: Rao Harish
Published: May 2021

Introduction

Overnight in-lab polysomnograms (PSG) are the gold standard for diagnosis of sleep disordered breathing in children. As the wait time for adenotonsillectomy (T & A) at our institution was several months, we implemented split night PSGs with positive airway pressure (PAP) during the initial diagnostic PSG if AHI>30 (Emergency SNPSG). Planned SNPSG were performed on children who are undergoing PSG after T & A, eliminating follow-up titration PSG if the PSG is positive for OSA (residual AHI>10/hour). We present data on the outcomes of the SNPSG.

Methods

Retrospective chart review of consecutive SNPSGs done over last 2 years at our institution was performed. Data on SNPSGs (planned or emergency), age, sex, diagnostic study duration, diagnostic study AHI, PAP pressure and subsequent PAP adherence were collected. Data on sleep efficiency, arousal, sleep architecture, REM sleep were compared between diagnostic and titration part of the SNPSG. Study was considered successful if patient was able to tolerate PAP during titration and also if adherent to PAP at follow up.

Results

48 studies met the criteria for SNPSG, with 60% of SNPSG being emergency SNPSG with AHI>30. Our cohort’s age ranged from 2–18 years (median age 8 years); 33 were males. Majority of the emergency SNPSG were in younger children (80% < 5 years), 75% of them continued to use PAP (mostly CPAP) until T & A with wait time being more than 3 months. Average wait time for T & A was 4 months. 25% of patients continued to use PAP following T & A as residual AHI was >10/hour. 98% of the patients were able to tolerate PAP during titration section with restoration of sleep architecture including REM with reduction in AHI, arousals and improved sleep efficiency. Bilevel PAP was used in 10% of patients in mostly planned SNPSG.

Conclusion

SNPSG can be implemented with fair degree of success during initial PSG with PAP used until T & A is performed. Planned SNPSG are also useful with residual severe OSA eliminating need for subsequent titration study. When indicated, 75% of our cohort continued to use PAP with fair adherence (>70% nightly use) following SNPSG.

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585 The Relationship between Napping and Behavioral Problems among Vocational High School Students in China
Authors: Han Jing , Huang Rendong , Yue Lei , Cui Naixue , Cao Fenglin
Published: May 2021

Introduction

Although napping is very common and related to the outcome of individual development, the relationship between napping and health is not the same in different social and cultural contexts. In China, napping is considered as a healthy lifestyle and is often associated with better adolescent development outcomes. As a special group of teenagers (the academic level does not meet the requirements of ordinary high school), vocational high school students have a higher incidence of behavioral problems than ordinary high school students. Therefore, the purpose of this study is to explore the characteristics of napping and its relationship with behavioral problems in vocational high school students.

Methods

The napping questionnaire, Youth Self Report (YSR), general information questionnaire and other tools for covariates were used to measure 2505 high school students (62.04% boys, 37.96% girls, 16.71±0.02 years), recruited by convenient sampling. The relationship between nappingand behavioral problems was analyzed by multiple linear regression.

Results

72.58% of the participants reported taking a midday nap at least three days per week during the past month, and 55.79% of our sample reported naps more than 30 minutes. Multiple regression analysis showed that nap frequency was negatively associated with high school students’ behavior problems after controlling for general characteristics and other important covariates. Compared with high school students who did not nap or napped less than 1 time/week, high school students who napped 1–2 times/week or 3–4 times/week had lower level of both internalizing behavior problems and externalizing behavior problems. There was no statistically significant association between nap duration and behavior problems.

Conclusion

This study finds that when napping is allowed, moderate frequency of napping is associated with lower level of internalizing and externalizing behavioral problems in vocational high school students, while nap duration is not significantly associated with behavioral problems. Further research is needed to explore the mechanism of the relationship between napping and behavior problems.

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586 Large Body Movements on Video-Polysomnography are Associated with Daytime Dysfunction in Children with Restless Sleep Disorder
Authors: Liu Wei , Thomas Dye , Horn Paul , Garner David , Simakajornboon Narong
Published: May 2021

Introduction

Restless sleep disorder (RSD) is a recently described disorder of children and adolescents with complaints of restless sleep and is characterized by large body movements which interfere with nocturnal sleep, are associated with daytime dysfunction, and are not better explained by another disorder (1, 2). Polysomnography criteria for RSD includes the scoring of large body movements (LBMI) and identified an LBMI ≥ 5 as sensitive and specific for RSD. As data is currently limited to two pediatric centers, our study aims to identify RSD cases at our institution, characterize their PSG, and determine their effect on daytime dysfunction and quality of life.

Methods

This single center retrospective study included 41 children with complaints of restless sleep and 35 sex- and age-matched children with RLS/PLMD collected from February 2018 to November 2020. The video PSGs were re-scored utilizing the previously published criteria (1,3). We used two LBMI thresholds, LBMI ≥ 5 and an exploratory LBMI ≥ 4. We then compared Epworth Sleepiness Scores (ESS), Pediatric Quality of Life measures (PedsQL), PSG characteristics, LBM associated awakenings and ferritin level between groups.

Results

Twenty-one children (mean age=8.3 ± 3.13 SD) met the LBMI>=4 criteria, of which 11 (age=8.4±3.81 SD) met the LBMI>=5 criteria. All three groups reported decreased quality of life (PedsQL total LBMI>=4=66.21±20.28, LBMI>=5=72.39±18.24, RLS/PLMD=55.54±19.86), low mean ferritin values (LBMI>=4=33.18 ± 20.11, LBMI>=5 =28.38 ± 12.68, RLS/PLMD = 36.57±25.46), and increased wake after sleep onset (WASO) (LBMI>=4 = 42.82 ± 34.26, LBMI>=5=44.38 ± 38.4, RLS/PLMD = 52.34 ± 42.03), with no significant differences between groups. Both LBMI>=4 and LBMI>=5 groups exhibited higher ESS compared to the RLS/PLMD group (LBMI>=4=10.36±7.13; LBMI>=5=11.13±5.19; RLS/PLMD=6.17±4.04; LBMI>=4 vs RLS/PLMD, P=0.05; LBMI>=5 vs RLS/PLMD, P = 0.032). Both awakenings and WASO associated with LBM were high in LBMI>=4 and LBMI>=5 (Awakenings: LBMI>=4=8.43±9.96, LBMI>=5 =8.00±12.19, WASOassociatedLBM: LBMI>=4=23.60±44.64 LBMI>=5=29.46±57.57)

Conclusion

One quarter of children with complaints of restless sleep met the standard criteria for RSD. Both children with RSD (including the exploratory LBMI≥ 4 group) and RLS/PLMD had increased WASO, low ferritin and decreased quality of life, but the RSD group had more daytime sleepiness compared to RLS/PLMD.

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588 Wake Up and Learn: A School Based Sleep Education and Surveillance Program
Authors: Morse Anne Marie , Blessing Kristina , Snyder Mallory , Liscum Denise
Published: May 2021

Introduction

Insufficient sleep, especially for students, has been a hot topic in news for the past few years. However, despite this increase in media attention, the number of adolescents with unrecognized sleep difficulties continues to grow. A partnership between Janet Weis Children’s Hospital (Geisinger) and Montgomery School District was established to pilot a program called “Wake Up and Learn” (WUAL). WUAL is a population based preventative sleep screening and education program for 7th to 12th graders through an asynchronous virtual platform. This is a descriptive summary of implementation of WUAL and early phase results.

Methods

The WUAL team consists of a board-certified pediatric sleep specialist, school guidance counselor and 2 project managers. The Protection of Pupil Rights Amendment (PPRA) was considered. A letter describing the program with an opt-out option was provided. WUAL website was developed to serve as an educational resource and to access the surveys. The surveys were generated using REDcap and included the Epworth sleepiness scale -CHAD (ESS) and the childhood sleep habits questionnaire (CHSQ). The surveys became available online and the students were instructed access the website and complete the surveys as part of class time.

Results

A total of 289 surveys were accessed, 287 ESS (99%) and 281 CSHQ (97%) were completed. Pathologic ESS scores (> 9), suggesting excessive daytime sleepiness (EDS), was identified in 75 students (26%). Scores suggestive of sleep dysfunction (> 41) on the CSHQ was found in 184 students (65%). Out of the 184 students 64 students also had an abnormal ESS.

Conclusion

This method of screening has demonstrated a high degree of successful completion. Based on early data, sleep pathology may be more prevalent than typically reported in children (20–25%). EDS was common, present in a quarter of students. However, of 184 students with abnormal CHSQ only 35% also had EDS. This suggests that there may be a need to identify alternative daytime dysfunction, such as school performance or mental health symptoms. This is an ongoing program that will have further updates and plans for expansion to more school districts.

Support (if any)

Grant Funding Support from Jazz Pharmaceuticals and Janet Weis Children’s Hospital (Geisinger)
616 Restless sleep and other comorbidities in children with attention deficit hyperactivity disorder
Authors: Kapoor Vidhi , Ferri Raffaele , Stein Mark , Ruth Chris , Reed Jennifer , Delrosso Lourdes
Published: May 2021

Introduction

The etiology of restless sleep has recently been identified as secondary to various medical and sleep conditions. Parents of children with ADHD complain of restless sleep and sleep disturbances in their children. We aimed to assess restless sleep causes in our patient population with ADHD and establish the prevalence of restless sleep disorder (RSD), as recently defined.

Methods

A retrospective study of children with ADHD who underwent polysomnography was carried out. Diagnostic and descriptive information collected for each patient included age, sex, polysomnographic parameters, sleep disorders, psychiatric comorbidities, and medications.

Results

Sixty-six PSGs were reviewed. The mean age of children was 11.6 (±3.6 SD) years; 17 were female and 49 were male. Parents of 54 (81.1%) children had concerns of restlessness during sleep; 47 (71.2%) children had obstructive sleep apnea, 17 (25.8%) had PLMS ≥5/hour, 13 (19.7%) had RLS, 6 (9.1%) had RSD, 27 (41%) had depression or anxiety, 5 (7.6%) had insomnia.

Conclusion

In a pediatric sleep medicine referred group of patients with ADHD, who are known to have significant sleep and psychiatric comorbidities, obstructive sleep apnea, RLS, and RSD were found to be the most prevalent sleep disorders, whereas depression and anxiety were the most common psychiatric disorders. Complaint of secondary restless sleep seems to be common, while primary restless sleep disorder was seen in approximately 9% of children. The results of this study aid pediatricians and child psychiatrists in screening children with ADHD for associated sleep disorders.

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629 Sleep problems among children and adolescents with epilepsy in Thailand: single-center cross-sectional study
Authors: Jaroenying Rasintra , Rattanarat Pluemkamon , Suwanpakdee Piradee
Published: May 2021

Introduction

Sleep problems are common in pediatric epilepsy and may influence seizure control, daytime performance, and quality of life. The aim of this study was to investigate parent-reported and self-reported sleep problems in a sample of children and adolescents with epilepsy in Thailand.

Methods

Epileptic children age 1–18 years and their parents in epilepsy clinic completed the Phramongkutklao Hospital Sleep disorders center Questionnaire, Sleep Related Breathing Disorder- Pediatric Sleep Questionnaire (SRBD-PSQ)

Results

A total of 59 participants with the mean age of 10.8 years completed the questionnaires. The most common sleep complaint was a disorder of maintaining sleep 36 (61.01%). Several sleep manifestations were detected including disorder of initiating sleep 14 (23.7%), snoring 29 (49.2%), leg kicking 16 (27.1%), sleep talking 19 (32.2%), sleep walking 4 (6.7%), teeth gliding 18 (30.5%), nightmares 15 (25%), night terrors 8 (13.5%) and nocturnal enuresis 16 (27.1%). The frequency of sleep disordered breathing problems (SRBD-PSQ >0.33) was 33.9%.

Conclusion

The prevalence of sleep problems among children with epilepsy in Thailand is high. Therefore, a highlighting should be given to address sleep in children and adolescents with epilepsy. Further studies are needed to evaluate the effects of antiepileptic drugs, type of seizure or associated factors on sleep disruption in this population.

Support (if any)

None