Journal of Clinical Sleep Medicine最新文献

This study presents two cases of central sleep apnea syndrome in children, highlighting the utility of assessing ventilatory control stability, particularly loop gain and central chemosensitivity in treatment decision-making. In the first case, elevated loop gain for oxygen correlated with periodic breathing, leading to successful treatment with supplemental oxygen in a 13 year-old boy with Prader-Willi-like syndrome. Conversely, in the second case, dealing with a 10 year-old girl with tumor in the brainstem-spinal cord junction, reduced loop gain prompted treatment with nocturnal non-invasive ventilation. These findings underscore the potential clinical relevance of loop gain measurement in pediatric central sleep apnea. While further research is needed to validate these findings in larger cohorts, loop gain endotyping shows promise as a tool for personalized treatment selection in pediatric sleep-disordered breathing.

Study objectives: Here we report our experience treating patients with narcolepsy using benzodiazepine receptor agonists (BzRA), zolpidem (Zol) or eszopiclone (Esz) taken at bedtime for both excessive daytime sleepiness (EDS) and cataplexy.

Methods: We reviewed the medical records of 53 patients diagnosed with narcolepsy, between 2002 and 2023. Twenty-three patients, 8 with type1 (NT1), 13 with type 2 (NT2) and 2 with secondary narcolepsy, were treated with BzRA's (20 Zol and 3 Esz).

Results: Seven out of 8 (88%) with NT1, 9 out of 13 (69%) with NT2 and 2 out of 2 (100%) with secondary narcolepsy, treated with BzRA, had good to excellent subjective response in their symptoms of EDS and/or cataplexy; 5 patients, 1 of whom with NT1, had marginal or no response. Three of the responding patients remained on zolpidem in monotherapy (ie. no stimulants).

Conclusions: The BzRa drugs may be effective to manage several of the cardinal symptoms of narcolepsy, regardless of the narcolepsy type. Placebo controlled trials are needed to confirm our observations.

Study objectives: This study examined the impact of central disorders of hypersomnolence (CDH) on family members of adult patients, the ways family members assist with managing CDH, and family members' utilization and satisfaction with information and support.

Methods: Participants were adults (N=100) with an adult family member diagnosed with idiopathic hypersomnia or narcolepsy. They completed a survey which included the Family Reported Outcome Measure (FROM-16), checklists, satisfaction ratings, and open-response questions.

Results: The FROM-16 sample mean (14.2, SD=6.8) corresponded to a moderate effect on quality of life. Compared to parents, partners reported a higher impact on the Personal and Social Life domain (p=.04, d=.44). The most frequently endorsed sources of support were family (60.0%) and friends (50.0%), whereas information was most commonly obtained from hypersomnia organizations (69.0%) and medical professionals (61.0%). Only 8.0% of participants were satisfied with support, and 9.0% were satisfied with information. Participants endorsed assisting with managing CDH, such as picking up prescriptions (61.0%), attending medical visits (50.0%), reminding to take medication (48.0%), and coordinating medical care (39.0%). Qualitative data indicated that relationships underwent a transformation from conflict and confusion (pre-diagnosis) to clarity (post-diagnosis), followed by adjusting expectations. Caregiving strain, effects on shared activities, and negative psychosocial impacts on family also emerged as themes.

Conclusions: Family members play an important role in supporting adults with CDH in many ways, including tasks related to managing CDH. Family members experience many psychosocial impacts from CDH, and data from this study indicate unmet needs for support.

Study objectives: Sleep disorders and/or disordered sleep represent common clinical presentations of pediatric acute-onset neuropsychiatric syndrome (PANS), occurring in up to 80% of affected children, with REM sleep motor disinhibition being a prevalent feature. To date, limited polysomnographic (PSG) studies have been conducted. Therefore, the objective of this study was to evaluate the PSG characteristics of a cohort of children with PANS, focusing particularly on REM sleep without atonia (RSWA) as assessed by the REM atonia index (RAI), and to compare these characteristics with those of a control group.

Methods: Sixty-nine patients diagnosed with PANS were consecutively recruited who underwent comprehensive PSG examinations following standard criteria. Chin muscle tone during REM sleep was evaluated using RAI. Forty-four healthy subjects, matched for age and sex, were used as controls.

Results: RAI was significantly lower in patients compared to controls. In controls, RAI showed a significant progressive increase with age from preschool to adolescence. Conversely, this age-related trend was absent in patients, with no significant correlation between RAI and age, with a random distribution of values. Only one control subject (1/43) but 25 patients (25/57) exhibited a RAI below the lower limit of the predicted range obtained from controls.

Conclusions: Our findings underscore the significance of REM sleep abnormalities in children with PANS. The substantial reduction in RAI and the lack of correlation between RAI and age observed in patients suggest a potential pathological mechanism leading to dysfunction in brainstem structures regulating REM sleep atonia in this population.

Study objectives: Obstructive sleep apnea (OSA) is common in Down syndrome (DS) with many patients prescribed positive airway pressure (PAP) therapy. This study evaluates PAP adherence and identifies factors influencing adherence.

Methods: Retrospective analysis of electronic health records and cloud-based PAP therapy data from DS patients at Rady Children's Hospital, San Diego, CA. Cloud data were evaluated cross-sectionally at 30- and 90-nights post-clinic visit and longitudinally in patients with ≥2 90-night data downloads. Outcomes included compliance (percentage of nights with ≥4 hours of use) and usage (percentage of nights with any PAP usage). The impact of demographic and PAP therapy factors (e.g., mask leak) on these outcomes was also assessed.

Results: 47 patients with DS with cloud-based PAP therapy data over a 90-night period and 46 over a 30-night period were analyzed. The mean age was 17.7±4.6 years (21 females). Median compliance was significantly higher at 30 nights (56.7%, IQR:0.0,90.8%) than at 90 nights (34.4%, IQR:0.0,86.7%) (p<0.05). Median usage did not differ between the 30-night and 90-night periods. Demographic characteristics and PAP therapy parameters were not associated with compliance or usage. Among the longitudinal cohort (n=32), median compliance was 69.7% (IQR:19.2,90.0%) and median usage was 78.2% (IQR:45.2,95.7%). Compared to an age- and sex-matched cohort without DS, patients with DS demonstrated higher PAP compliance (p<0.05).

Conclusions: Cross-sectional and longitudinal analyses reveal that many DS patients successfully adhere to PAP therapy, challenging the misconception that they struggle with adherence and proving they may be as successful, if not more, than non-DS patients.

Study objectives: The prevalence of obstructive sleep apnea (OSA) increases dramatically in adolescents with overweight or obesity. The gold standard for diagnosis of OSA is in-laboratory polysomnography (PSG). However, access to PSG can be challenging, necessitating development of alternative devices. This study aimed to compare the accuracy of WatchPAT, a device that indirectly detects apnea and hypopnea events through peripheral arterial tonometry, to PSG in adolescents with overweight and obesity.

Methods: 38 participants (15.5±1.6 years; 76% female; 40% Hispanic/Latino; BMI 97.7±2.0 percentile) were analyzed. Correlation, agreement, sensitivity, specificity, and concordance between WatchPAT with PSG-derived measures of apnea-hypopnea index (AHI) and OSA severity were conducted. A subset (n=21) underwent subjective assessment of nasal flow sensor reliability and video characteristics to evaluate possible underestimation of PSG-assessed AHI.

Results: Mean bias between WatchPAT- and PSG-derived AHI was 16.9±13.4 events/hour (95% CI 12.5-21.3). WatchPAT overestimated OSA severity compared to PSG in 89% of participants. Sensitivity was 62-100% and specificity was 5.5-32% across all OSA severity levels. Of the subset with subjective assessment of PSG data, 38% had nasal flow sensor reliability <75%, 14% had underestimated AHI, and 28% had probably underestimated AHI. However, these characteristics did not fully explain the discrepancy between WatchPAT and PSG measurements.

Conclusions: The WatchPAT device showed a significant discrepancy compared to the gold-standard PSG in measurement of AHI and accuracy of OSA severity in adolescents with overweight or obesity. Future research is needed to understand pathophysiological differences to enhance assessment of OSA in this high-risk population.

Study objectives: Obstructive sleep apnea (OSA) is a common disorder in the pediatric population, primarily diagnosed through polysomnography (PSG). However, PSG can be expensive and is often limited in availability. This study aimed to develop a cost-effective diagnostic strategy by integrating a screening model with PSG.

Methods: A retrospective analysis was conducted on children suspected of OSA. Screening models were initially constructed with machine learning techniques. Cost-utility analyses compared three diagnostic strategies: (1) PSG alone, (2) the screening model alone, and (3) the screening model-PSG combined, in the discovery and validation cohorts. Cost-utility was measured using the incremental net monetary benefit (INMB).

Results: 690 children were included. The logistic regression model using age, tonsil scale, OSA-18 questions 1 and 2, and oxygen desaturation index 3% predicted OSA with an area under the curve of 0.91. In the cost-utility analysis, the "PSG alone" strategy, as the baseline, was the most beneficial (utility 0.9557) at Chinese Yuan (CNY) 4523.98. The "screening model alone" had 91.6% sensitivity and 59.3% specificity, offering lesser value (utility 0.9337) at CNY 6071.51 (INMB CNY -3966.43) when compared to "PSG alone". The "screening model-PSG combined" strategy increased sensitivity to 100%, specificity to 99.2%, and utility to 0.9554 at CNY 4463.36, establishing it as the most cost-effective option with an INMB of CNY 34.22. One-way sensitivity analyses and adaptation to US cost parameters confirmed the robustness of these results.

Conclusions: Using the screening model as a triage tool for PSG enhances the cost-effectiveness of pediatric OSA management.