bluuebunny/crossref_metadata_2025_split huggingface dataset:
524 NAPS
Authors: Elliott Jonathan , Lim Miranda , Keil Allison , Avidan Alon , Bliwise Donald , Gagnon Jean-Francois , Howell Michael , Huddleston Daniel , McCleland Jennifer , St. Louis Erik , Postuma Ronald , Videnovic Aleksandar , Boeve Bradley , Ju Yo-El 
Published: May 2021

Abstract

Introduction
Rapid Eye Movement (REM) Sleep Behavior Disorder (RBD) is characterized by a lack of muscle atonia during REM sleep with dream enactment. RBD is regarded as a prodromal synucleinopathy as a high proportion of patients eventually phenoconvert to Parkinson’s Disease and related synucleinopathies, suggesting RBD may be an early non-motor symptom of disease. Accordingly, patients with RBD are ideally situated to test potential therapeutic interventions to prevent phenoconversion to synucleinopathy. However, RBD itself, and associated patient registries, are rare. The North American Prodromal Synucleinopathy Consortium (formed in 2018) establishes a multisite registry of RBD patients with standardized neurological, neuropsychiatric, and neuropsychological assessments and biomarker collection. The present work reports baseline characteristics of this RBD patient database at its current state.

Methods
Seven participating sites have contributed n=170 polysomnographically-confirmed RBD patients. Data includes past medical and family history, self-report questionnaires, and neuropsychological, motor, sensory, and autonomic function testing. Additionally, all subjects have contributed blood, and a subset of subjects have contributed cerebrospinal fluid samples to the National Centralized Repository for Alzheimer’s Disease and Related Dementias for future analysis. A final diagnosis for each subject was determined through an adjudication process by NAPS Consortium PIs; subjects were categorized as ether: 1) isolated RBD, 2) RBD+, 3) Early Symptomatic, or 4) Phenoconverted.

Results
Of the n=170 subjects, there were n=39 isolated RBD, n=81 RBD+, n=45 Early Symptomatic, and n=4 Phenoconverted. Isolated RBD subjects have no other early neurodegeneration signs/symptoms, those with RBD+ have at least one other identifiable early/mild symptom. The early symptomatic group includes those with mild or subjective cognitive impairment, pure autonomic failure, or possible multiple systems atrophy. The Phenoconverted group consists of those with Dementia with Lew Bodies, Dementia NOS, Parkinson’s Disease, or Parkinson’s NOS. The distribution of impairment across the 5 major domains (motor, cognitive, autonomic, sensory, and psychiatric) for each of the 4 groups will be described.

Conclusion
This interim analysis presents data on n=170 subjects. The target enrollment is n=360 across the 7 original sites plus 3 new sites. Future work will follow these subjects longitudinally to assess rates and predictors of phenoconversion.

Support (if any)
NIH NIA R34 AG056639 (YJ, BB)
525 A population-based study of phenoconversion to parkinsonism from REM sleep behavior: a Population-based Study in the CLSA
Authors: Yao Chun , Zolfaghari Sheida , Chaudhuri Paramita Saha , Pelletier Amelie , Wolfson Christina , Postuma Ronald 
Published: May 2021

Abstract

Introduction
To date, studies have estimated the phenoconversion rate from sleep clinics, using polysomnography proven RBD. However, no population-based estimates have been reported, testing to what degree possible RBD, screened by questionnaire is associated with increased risk of neurodegeneration.

Methods
We included those aged 45–85 years, living in one of 10 Canadian provinces in between 2012–2015 (at the baseline), recruited via three population-based sampling methods. Dream enactment behavior/possible RBD was screened using the RBD1Q single question-questionnaire. De-novo parkinsonism was defined as free of pre-existing diagnosis at the baseline with a ‘new’ diagnosis at the follow-up (205–2019). Relative risk (log-binomial regression), hazard ratio (Cox regression), incidence rate (Poisson regression) between the affected group and the symptom naïve group were assessed, adjusting for age and sex (and total years of education and language).

Results
Overall, 58 participants phenoconverted into parkinsonism and 53 into dementia at the follow-up (mean intervals=3.06±0.37 years). Participants with dream enactment behavior had 2.75 times higher risk to phenoconvert into parkinsonism than the symptom-free. Similarly, those with dream enactment behavior at the baseline possessed higher risk to screening positive of parkinsonism. No difference in time to phenoconversion was found between groups, The results remained robust after excluding non-RBD related symptoms, such as apnea and non-REM sleep parasomnia.

Conclusion
Compared to symptom-free, those with pRBD had higher risk to developing parkinsonism in near future.
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533 Characteristics of patients with REM sleep without atonia (RSWA)/persistent periodic limb movements of sleep (PLMS) in REM sleep
Authors: Barker Lina , Tippmann-Peikert Maja 
Published: May 2021

Abstract

Introduction
While RSWA in REM sleep behavior disorder (RBD) is associated with male sex, age &gt; 50 years, alpha-synucleinopathies, and narcolepsy, the characteristics of patients with RSWA/PLMS in REM sleep (RSWA/PLMS-REM) without dream enactment behaviors are unexplored. The aim of this study was to compare the demographics, comorbidities and concomitant medication use between RSWA/PLMS-REM patients and non-RSWA/non-PLMS-REM controls. Based on anecdotal clinical observations we hypothesized that these patients are more commonly young, women, have psychiatric or neurological diseases, and use antidepressants.

Methods
We conducted a retrospective review of the Mayo Clinic electronic medical record to identify all patients with RSWA/ PLMS-REM between November 2018 and November 2020. After excluding all patients with RBD, restless legs syndrome, narcolepsy and RSWA/non-PLMS-REM we identified 27 patients. All PSGs were reviewed to calculate the periodic limb movement index per hour of REM sleep (REM-PLMI). We also identified a covenience sample of 15 non-RSWA controls, reviewed their PSGs and calculated REM-PLMI.

Results
The mean REM-PLMI of patients with RSWA was 64+/-8.3 (SEM)/hour versus 1+/-0.6 (SEM)/hours in non-RSWA controls (p&lt; 0.001). Patients with RSWA/PLMS-REM and non-RSWA controls had similar age and gender, 62 +/- 3 (SEM) versus 58 +/-3 (SEM) years and 81% versus 87% men, respectively. However psychiatric diagnosis, neurological disorders and antidepressants use was more common among RSWA/PLMS-REM patients compared to non-RSWA controls, p=0.0002, p=0.0035 and p=0.0074, respectively. (Fisher’s Exact Test)

Conclusion
Psychiatric diagnosis, neurological disorders and antidepressant use are more common among RSWA/PLM-REM patients compared to non-RSWA controls. Further research to determine the implications of a diagnosis of RSWA/PLMS-REM for the future development of alpha-synucleinopathies are needed and currently ongoing.
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540 Age Trends in Sleep Across the Lifespan: Findings from the Pittsburgh Lifespan Sleep Databank
Authors: Wallace Meredith , Kissel Nicholas , Hall Martica , Germain Anne , Matthews Karen , Troxel Wendy , Franzen Peter , Buysse Daniel , Roecklein Kathryn , Gunn Heather , Hasler Brant , Goldstein Tina , McMakin Dana , Szigethy Eva , Soehner Adriane 
Published: May 2021

Abstract

Introduction
Sleep continuously changes over the human lifespan and it does so across multiple dimensions, including duration, timing, efficiency, and variability. Although studies focused on specific developmental periods have shown age-related changes in sleep, methodological differences make it difficult to synthesize information across studies to fully understand precisely when these sleep changes occur. Our goal was to use individual-level actigraphy and self-report sleep data from a single site to characterize age trends and sex differences in actigraphy and self-report sleep dimensions across the healthy human lifespan. To accomplish this goal, we developed the Pittsburgh Lifespan Sleep Databank (PLSD), a large aggregate databank of participants from sleep research studies conducted at the University of Pittsburgh.

Methods
In the present analysis, we included N=1,070 PLSD participants from 21 studies without a major psychiatric, sleep, or medical condition. We used Generalized Additive Models to examine flexible, potentially non-linear relationships between age and sleep dimensions (actigraphy and self-report duration, efficiency, and timing; actigraphy variability) from ages 10 to 87. We also examined whether these sleep characteristics differed by sex across the lifespan.

Results
The most dramatic age-related trends were observed in sleep timing. Actigraphy and self-report sleep onset time shifted later between ages 10–18 and then shifted earlier again during the 20s. Actigraphy and self-report wake-up time also shifted earlier during the mid-20s through late 30s. Self-report duration became shorter from approximately ages 10–20. Self-report sleep efficiency and actigraphy variability both decreased over the entire lifespan. Relative to males, females tended to have earlier self-report sleep onset, higher actigraphy sleep efficiency, and longer actigraphy duration.

Conclusion
By focusing on lifespan sleep rather than specific age segments of the samples, we can provide a unified assessment of age-related changes and sex differences from childhood through older adulthood. An understanding of age trends and sex differences in sleep in healthy individuals – and explicating the timing and nature of these difference – can be used to identify periods of sleep-related risk or resilience and guide intervention efforts.

Support (if any)
University of Pittsburgh Clinical and Translational Science Institute (UL1TR001857).
541 Cognitive Hyperarousal, Sleep Problems and Temperament Linked to Impaired Maternal-Infant Bonding
Authors: Arnett Lily , Kalmbach David , O’Brien Louise , Pitts D’Angela , Harb Nicholas , Drake Christopher 
Published: May 2021

Abstract

Introduction
Emerging evidence links maternal and infant sleep problems to impairments in the mother-to-infant bond, but the independence and directionality of these associations remain unclear. The present study characterized concurrent and prospective effects of maternal sleep disturbances and poor infant sleep on the mother-infant relationship. As common sequalae of problematic sleep, nocturnal cognitive hyperarousal and daytime sleepiness were investigated as facilitating mechanisms.

Methods
Sociodemographic information and clinical symptoms were measured prenatally then weekly across the first two postpartum months in 67 women. Participants reported insomnia symptoms, sleep duration, snoring, daytime sleepiness, nocturnal cognitive arousal (broadly focused and perinatal-specific), perseverative thinking, depression, infant colic, infant sleep quality, and mother-infant relationship quality. Mixed effects models were conducted to test hypotheses.

Results
Prenatal snoring and weak maternal-fetal attachment augured poorer postpartum bonding. Poor infant sleep was associated with increased odds for maternal insomnia and short sleep. Impairments in the mother-to-infant bond were linked to maternal insomnia, nocturnal perinatal-focused rumination, daytime sleepiness, depression, and poor infant sleep. Postnatal insomnia predicted future decreases in mother-infant relationship quality, and nocturnal cognitive hyperarousal partially mediated this association.

Conclusion
Both maternal and infant sleep problems were associated with impairments in mother-to-infant bonding, independent of the effects of maternal depression and difficult infant temperament. Perseverative thinking at night, particularly on infant-related concerns, was linked to impaired bonding, rejection and anger, and infant-focused anxiety. Improving maternal and infant sleep, as well as maternal cognitive-emotional regulation, may improve the maternal-to-infant bond.

Support (if any)
This work was funded by the American Academy of Sleep Medicine (198-FP-18, PI: Kalmbach). Dr. Cheng’s effort was supported by the National Heart, Lung, and Blood Institute (K23 HL138166, PI: Cheng).
548 Predicting Perceived Functional Limitation in Midlife and Older Adulthood: The Role of Sleep and Perceived Control
Authors: Williams Claire , Ghose Sarah , Reid Morgan , Sabet Sahar , MacPherson Ashley , Dautovich Natalie , Dzierzewski Joseph 
Published: May 2021

Abstract

Introduction
Functional limitations become increasingly common and debilitating as individuals age, potentially impacting several facets of well-being. As such, it is important to understand malleable factors that may potentially impact functional limitation outcomes. Both sleep and perceived control have been linked to the development of functional limitation. The current study sought to clarify the unique contributions of both sleep quality and perceived control to functional limitation status in middle-aged and older adults.

Methods
Data from the second wave of the Midlife in the United States study were used for the current study. Participants included 527 participants (59.9% female, Mage=59.83 years, SD=9.75 years) who completed measures of functional limitation levels (Functional Status Questionnaire), subjective sleep quality (Pittsburg Sleep Quality Index), and perceived control (MIDI Sense of Control Scales). A hierarchical regression analysis was conducted to examine sleep quality and perceived control as predictors in a unique model for predicting functional limitation. Demographic variables of age, gender, and race were used as covariates in study analyses.

Results
The overall model predicted 19.0% of the variance in functional limitation levels. Sleep quality was significantly associated with self-reported functional limitation (β=-.27, p&lt;.001) over and above perceived control (β=.20, p&lt;.001). Specifically, findings indicate that worse sleep quality is associated with increased functional limitation, while higher levels of perceived control are associated with lower levels of functional limitation.

Conclusion
Though perceived control is known to be associated with functional limitation status, the present study suggests a unique effect of sleep quality on functional limitation even after accounting for perceived control. Due to the potential for negative effects of functional limitation in middle-aged to older adults, it is important to identify and target constructs for research and intervention related to the development of these limitations. Care models for individuals who report experiencing functional limitations may benefit from targeting sleep health and control beliefs in intervention and assessment.
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550 Laryngomalacia and obstructive sleep apnea in infants with PWS
Authors: Powell Weston , Wrede Joanna , Salehi Parisa 
Published: May 2021

Abstract

Introduction
Growth hormone (GH) improves tone, body composition, development, and growth in infants with Prader-Willi Syndrome (PWS). Concerns about sudden death following GH initiation and worsening obstructive sleep apnea (OSA) in children with PWS resulted in guidelines for polysomnography (PSG) evaluation before and after starting GH. We review novel evidence of laryngomalacia as a mechanism for this worsening of OSA and describe the incidence of laryngomalacia in this patient population.

Methods
A retrospective review of infants and children seen at the Seattle Children’s PWS clinic between October 2014 and May 2020 who had undergone polysomnography (PSG) before and after growth hormone initiation was performed. Findings on otolaryngology evaluation via flexible fiberoptic laryngoscopy (FFL) or drug-induced sleep endoscopy were reviewed to characterize obstruction, diagnosis of laryngomalacia, and response to surgical intervention.

Results
A total of 28 cases were identified. 12 (41%) were evaluated with FFL between ages 4 and 21 months old (median 5) for noisy breathing, worsening or persistent OSA, or dysphagia. Out of these, 9 (75% of FFL, 31% of total) were diagnosed with laryngomalacia. Children with laryngomalacia were more likely to have worsening of OSA after GH initiation. Surgical interventions including supraglottoplasty or adenotonsillectomy led to improvement in OSA in 86% of children who had worsening after GH initiation.

Conclusion
Worsening OSA after GH initiation is seen in 38% of patients with PWS. Laryngomalacia is a common comorbid condition and more frequent in those with worsening OSA after GH initiation that is amenable to targeted surgical intervention.
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552 Impact of Adenotonsillectomy on Growth Trajectories in Preschool Children with Mild Obstructive Sleep Apnea
Authors: Kevat Ajay , Chawla Jasneek , Bernard Anne , Harris Margaret-Anne , Heussler Helen , Black Robert , Waters Karen 
Published: May 2021

Abstract

Introduction
Adenotonsillectomy forms part of first-line management for pediatric obstructive sleep apnea (OSA). In nonrandomized studies of preschool-aged children, it is associated with postoperative weight gain. Being overweight or obese in childhood is a predictor of cardiovascular and metabolic disease in later life. Using longitudinal data from a multicenter randomised controlled trial, we assessed the impact of adenotonsillectomy on growth trajectory in preschool-aged children with mild-moderate OSA. Secondary aims were to assess the influence of social factors and baseline polysomnography parameters on growth trajectory.

Methods
A total of 190 children (aged 3–5 years) with obstructive apnea hypopnea index ≤10 were randomly assigned to early (within 2 months) or routine (12-month wait) adenotonsillectomy. Anthropometry and polysomnography were performed at baseline, 12-month and 24-month timepoints for 126 children. Social risk factors were recorded using a questionnaire. Baseline characteristics were compared using a Mann-Whitney or t-test for continuous variables, and Fisher’s exact test for categorical variables. Data were analyzed using linear mixed modelling.

Results
Demographic and polysomnographic parameters were similar between groups at baseline. Baseline body mass index (BMI) z-score was 0.52 for both groups. For BMI z-score, there was a significant increase in the early surgery group between 0 and 12 months (0.4, 95%CI 0.1–0.8) but not from 12–24 months. For the routine surgery group, there was a significant BMI z-score increase following surgery between 12 and 24 months (0.45, 95%CI 0.1–0.8), but not from 0–12 months. Final BMI z-score was similar between the two groups. Findings for weight-for-age z-score were similar to the abovementioned findings for BMI z-score. Height-for-age z-score was not significantly different between different timepoints or intervention groups. Children with an unemployed primary income earner had a higher BMI z-score than those with a full-time employed income earner. No other social risk or polysomnography parameters were statistically significant.

Conclusion
This study provides randomized controlled trial evidence of notable weight increase in preschool children with milder spectrum OSA that occurs in the months immediately following adenotonsillectomy. For children undergoing adenotonsillectomy, counselling regarding nutritional intake and exercise alongside weight monitoring should be considered, especially for those already at risk of becoming overweight or obese.
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556 Lost to Follow-Up: Post-Operative Polysomnography in High-Risk, Pediatric OSAs
Authors: Long Benjamin , Arora Subodh , McKinlay Alex , Foster Shannon , Hansen Shana 
Published: May 2021

Abstract

Introduction
Post-operative polysomnography (PSG) is recommended in certain pediatric populations at risk for residual sleep disordered breathing: moderate to severe obstructive sleep apnea syndrome (OSAS), obesity, craniofacial abnormalities, and neurologic disorders. In light of multiple stakeholders involved in follow-up management, variability in completion of a post-operative PSG may exist. We hypothesize that patients with isolated severe OSAS or severe plus a co-morbidity will have greater incidence of a post-operative PSG.

Methods
A chart review of 373 pediatric patients revealed 67 patients who met inclusion criteria for our high-risk cohort. Chart review included the presence of an ENT, Primary Care, or Sleep Medicine encounter, time to follow-up, the presence of a post-operative PSG, time to post-operative PSG, and the presence of an annual follow-up with any provider.

Results
Although 83% of our cohort followed-up with any provider, only 31% completed a post-operative PSG. Patients consistently followed-up with ENT 6–8 weeks postoperatively (76%) and haphazardly followed-up with primary care (38%). All patients with a Sleep Medicine follow-up (19%, n=13) completed a post-operative PSG, with 11 of the 13 occurring within 1 year from surgery. There was no significant difference across isolated moderate, isolated severe, or moderate/severe with a comorbidity for incidence of follow-up by specialty, annual follow-up, or post-operative PSG completion. However, patients with isolated severe (AHI &gt;10) completed a PSG on average 13.5 weeks post-operatively which was significantly sooner than 36.2 weeks for isolated moderate OSA (p=0.04).

Conclusion
Although Sleep Medicine providers may consistently follow AASM practice parameters, variability exists for which patients return to complete a post-operative PSG. Severity of OSAS or presence of a concerning co-morbidity does not seem to correlate with acquiring a postoperative PSG. An inconsistent standard across disciplines may contribute to this discrepancy. These findings will inform future quality improvement discussions with key stakeholders. In light of this baseline assessment, we plan to recommend a standardized, multi-disciplinary care pathway for the management of high-risk, pediatric OSAS.

Support (if any)
None