Item response theory analysis of daytime sleepiness as a symptom of obstructive sleep apnea.

Obstructive sleep apnea (OSA) is a sleep disorder which is linked to many health risks. The gold standard to evaluate OSA in clinical trials is the Apnea-Hypopnea Index (AHI). However, it is time-consuming, costly, and disregards aspects such as quality of life. Therefore, it is of interest to use patient-reported outcomes like the Epworth Sleepiness Scale (ESS), which measures daytime sleepiness, as surrogate end points. We investigate the link between AHI and ESS, via item response theory (IRT) modeling. Through the developed IRT model it was identified that AHI and ESS are not correlated to any high degree and probably not measuring the same sleepiness construct. No covariate relationships of clinical relevance were found. This suggests that ESS is a poor choice as an end point for clinical development if treatment is targeted at improving AHI, and especially so in a mild OSA patient group.

Clinical features and genetic spectrum of a multicenter Chinese cohort with myotonic dystrophy type 1.

BACKGROUND: As the most common subtype of adult muscular dystrophy worldwide, large cohort reports on myotonic dystrophy type I (DM1) in China are still lacking. This study aims to analyze the genetic and clinical characteristics of Chinese Han DM1 patients. METHODS: Based on the multicenter collaborating effort of the Pan-Yangtze River Delta Alliance for Neuromuscular Disorders, patients with suspected clinical diagnoses of DM1 were genetically confirmed from January 2020 to April 2023. Peak CTG repeats in the DMPK gene were analyzed using triplet repeat-primed PCR (TP-PCR) and flanking PCR. Time-to-event analysis of onset age in females and males was performed. Additionally, detailed clinical features and longitudinal changes from the disease onset in 64 DM1 patients were retrospectively collected and analyzed. The Epworth Sleepiness Scale and Fatigue Severity Scale were used to quantify the severity of daytime sleepiness and fatigue. RESULTS: Among the 211 genetically confirmed DM1 patients, the mean age at diagnosis was 40.9 ± 12.2 (range: 12-74) with a male-to-female ratio of 124:87. The average size of CTG repeats was 511.3 (range: 92-1945). Among the DM1 patients with comprehensive clinical data (n = 64, mean age 41.0 ± 12.0), the age at onset was significantly earlier in males than in females (4.8 years earlier, p = 0.026). Muscle weakness (92.2%), myotonia (85.9%), and fatigue (73.4%) were the most prevalent clinical features. The predominant involved muscles at onset are hands (weakness or myotonia) (52.6%) and legs (walking disability) (42.1%). Of them, 70.3% of patients had daytime sleepiness, 14.1% had cataract surgery, 7.8% used wheelchairs, 4.7% required ventilatory support, and 1.6% required gastric tubes. Regarding the comorbidities, 4.7% of patients had tumors, 17.2% had diabetes, 23.4% had dyspnea, 28.1% had intermittent insomnia, 43.8% experienced dysphagia, and 25% exhibited cognitive impairment. Chinese patients exhibited smaller size of CTG repeats (468 ± 139) than those reported in Italy (613 ± 623), the US (629 ± 386), and Japan (625 [302, 1047]), and milder phenotypes with less multisystem involvement. CONCLUSION: The Chinese Han DM1 patients presented milder phenotypes compared to their Caucasian and Japanese counterparts. A male predominance and an early age of onset were identified in male Chinese Han DM1 patients.

Making sense of narcolepsy: A qualitative exploration of how persons with narcolepsy perceive symptoms and their illness experience.

INTRODUCTION: Understanding how persons with narcolepsy conceptualize symptoms, daily impact and illness experience is key to facilitating dialogue between patients and healthcare professionals. These concepts are usually explored from the perspective of healthcare professionals/researchers and rarely from the perspective of those with narcolepsy. METHODS: 127 self-reported persons with narcolepsy were recruited from an Australian patient support group. A short demographic survey was completed. All agreed to participate in a subsequent 1:1 semi-structured interview. Saturation was reached after 24 interviews (mean age = 33 years (SD 11) with 44% reporting cataplexy). A multidisciplinary team of researchers/clinicians analyzed interview transcripts using thematic analysis. RESULTS: Participants perceived physical fatigue, sleepiness, and two separate experiences of 'falling asleep/sleep attacks' as distinct symptoms rather than a multidimensional construct (i.e. excessive daytime sleepiness). We also identified two experiences of cataplexy, one triggered by acute emotion and another by a stressor. Participants determined their narcolepsy to be 'well-managed' by the level of functional impairment rather than the frequency of any symptom. Almost all participants described experiencing anticipated stigma and internalized or 'self-' stigma, likely stemming from societal devaluation of sleep and the conflation of sleepiness with laziness. CONCLUSION: Descriptions of common symptoms often differed between participants and the existing literature. These differences likely impact patient-physician communication, with both parties utilizing the same terminology to communicate different concepts. The characterization of stigma in narcolepsy presents opportunities for future research exploring the impact and possible development of interventions to reduce the substantial psychological comorbidity in persons with narcolepsy.

Effects of childhood obstructive sleep apnea with and without daytime sleepiness on behaviors and emotions.

OBJECTIVE: To investigate the relationship between obstructive sleep apnea (OSA) with and without excessive daytime sleepiness (EDS) and behavioral and emotional outcomes in non-obese prepubertal children. METHODS: This was a retrospective analysis of children aged 5-11 years who presented to our unit for assessment of their sleep-related complaints. All children underwent polysomnography (PSG). They also completed the Pediatric Daytime Sleepiness Scale (PDSS) and a sleep diary. OSA was diagnosed if the obstructive apnea-hypopnea index (OAHI) was ≥1 event/hour. EDS was defined as PDSS >15. Behavioral and emotional outcomes were assessed using the Child Behavioral Checklist (CBCL). RESULTS: Data from 391 children (mean age of 8.6 ± 1.7 years; 67 % male) were analyzed. Seventy children did not have OSA or EDS, 137 had OSA, 50 had reported having EDS but without OSA, and 134 children had both OSA and EDS. There were significantly higher CBCL total problems score in the combined group (61 ± 9) compared to the non-OSA/EDS group (54 ± 10), and the OSA-only group (54 ± 10) (p < 0.001). The presence of EDS was significantly associated with higher CBCL T score and higher odds for clinically significant behavioral problems (T score ≥65) after adjusting for age, sex, BMI z-score and average sleep duration (p < 0.001). CONCLUSION: Excessive daytime sleepiness is an important contributory factor associated with suboptimal behavioral and emotional outcomes in children with OSA.

Self-reported sleep tendency poorly predicts the presence of obstructive sleep apnea in commercial truck drivers.

BACKGROUND AND OBJECTIVES: Drowsy driving increases the risk of motor vehicle crashes in those with untreated obstructive sleep apnea (OSA). Although previous studies indicated that excessive daytime sleepiness (EDS) might not predict OSA, they were not conclusive due to their small study sizes or restricted participants to sleep clinic patients. The overall objective was to determine whether self-reported EDS can be used for case identification of OSA among commercial truck drivers. METHODS: Commercial truck drivers (N = 19,699) were screened for OSA-related symptoms. EDS was determined using the Epworth Sleepiness Scale (ESS) ≥ 11 and all participants completed the home sleep apnea test using a type 4 portable monitor to derive the respiratory event index (REI). Regression analyses were used to characterize the association between EDS and REI. RESULTS: EDS was associated with OSA severity (p for trend <0.001). The sensitivity and specificity values of EDS for identifying moderate-to-severe OSA (REI ≥15 events/hour) were 0.10 and 0.93, respectively, and 0.48 and 0.71 if BMI ≥25 kg/m2 was added. Those using BMI ≥25 kg/m2 with OSA-related signs yielded the best sensitivity and specificity of 0.77 and 0.50, which were not improved by the addition of EDS. CONCLUSIONS: Despite the associations between EDS and OSA severity and between OSA and lethal crash, case-identification of OSA using the ESS in commercial truck drivers is poor. Thus, OSA screening strategy may need a special approach, including a hierarchical combination of screening tools (Swiss Cheese Model approach), and incorporation of home sleep apnea testing.

Supplement use and its impact on daily life of cancer patients: a cross-sectional study.

PURPOSE: This cross-sectional study aimed to investigate the prevalence and characteristics of supplement usage among cancer patients and explore its potential associations with anxiety, excessive daytime sleepiness, and overall quality of life. METHODS: Cancer patients receiving specific care at Hôtel Dieu de France University Hospital, Beirut, were enrolled between April and June 2023. In face-to-face interviews, participants were asked to complete a questionnaire consisting of sociodemographic information, supplement usage details, and cancer-related variables. Three validated surveys (Epworth Sleepiness Scale, GAD-7, and EORTC-QLQ-C15-PAL) were employed to assess excessive daytime sleepiness, anxiety, and overall quality of life. Statistical analyses, including chi-square tests, t-tests, and multiple regression models, were conducted to examine associations between supplement use and other variables. RESULTS: A total of 202 participants were interviewed. Fifty-two percent reported regular use of supplements following their cancer diagnosis, with vitamin D being the most commonly used supplement. Using multivariate logistic regression, supplement use was associated with being female, having lower educational levels, having a longer duration since cancer diagnosis, and having a poor overall quality of life. The multivariate logistic regression showed no significant correlation between supplement use and excessive daytime sleepiness and anxiety. CONCLUSION: This study highlights a high prevalence of supplement usage among cancer patients in Lebanon, indicating a rising interest in alternative therapies aimed at enhancing quality of life. Larger prospective studies are needed to assess the relation between supplement intake and excessive daytime sleepiness and anxiety and establish clear guidelines pertaining to supplement use in cancer patients.

Major depressive disorder with hypersomnolence complaint: A comparison study with non-depressed individuals examining objective biomarkers.

BACKGROUND: Hypersomnolence is common in major depressive disorder (MDD), associated with more severe episodes, suicide and antidepressant resistance. Nevertheless, few studies used polysomnography (PSG) and multiple sleep latency test (MSLT) to characterize these patients. In this context, we compared patients visiting a sleep center for hypersomnolence complaint with MDD (HSC/MDD+) and without MDD (HSC/MDD-). METHODS: HSC/MDD+ and HSC/MDD- groups were defined according to DSM-5 criteria and CES-D scale, and had a 30 h-PSG with ad libitum-sleep and PSG followed by MLST. RESULTS: HSC/MDD+ had an increased self-declared total sleep time (sTST) of about 10 h30 similar to HSC/MDD- (630.8 ± 17.3 min-vs-616.5 ± 18.1 min, respectively, p = 0.39). Nevertheless, their objective TST (oTST) on ad libitum PSG was significantly longer and about 10 h50 (648.6 ± 23.9 min-vs-587.4 ± 19.0 min, respectively, p = 0.038). HSC/MDD+ also significantly better estimated their sleep duration, with a lower difference between their sTST and oTST compared to HSC/MDD- (10.0 ± 1.7 %-vs-17.4 ± 2.1 %, respectively, p = 0.009) and confirmed significantly more frequently the hypersomnia diagnosis -i.e. oTST>10H- (82.6 ± 8.1 %-vs-54.6 ± 10.9 %, respectively, p = 0.046). Using the Kupfer index (KI), we confirmed a reduced REM sleep latency in patients MDD/HSC+ (15.2 ± 10.0 %-vs-2.3 ± 2.3 %, respectively, p = 0.039). Both groups had comparable increased diurnal sleepiness assessed with the Epworth scale (14.1 ± 1.1-vs-14.8 ± 1.1, respectively, p = 0.65). HSC/MDD+ had less MSLT sleep latency <8 min (9.1 ± 5.1 %-vs-27.3 ± 6.8 %, respectively, p = 0.048). LIMITATIONS: Retrospective cross-sectional study. CONCLUSIONS: HSC/MDD+ accurately estimated their sleep duration, objectively confirmed hypersomnia and may specifically had a decreased Kupfer index.

Idiopathic hypersomnia with a video recording of a spontaneous sleep attack: A case report.

RATIONALE: Although patients with central disorders of hypersomnolence (CDH) exhibit characteristic symptoms of hypersomnia frequently, it takes 5 to 15 years from the onset for its diagnosis due to the lack of symptom recognition. Here, we present a case of idiopathic hypersomnia (IH), a CDH, wherein early diagnosis was aided by a video footage of a spontaneous sleep attack. PATIENT CONCERNS: A 21-year-old man lost consciousness while driving and experienced an accident. He had complained of excessive daytime sleepiness (EDS) over half a year. During his hospitalization for close monitoring of the loss of consciousness, an in-room surveillance camera captured a 14-minutes long spontaneous sleep attack, during which he experienced general muscle weakness and loss of consciousness without warnings or convulsions leading to a fall from the bed. There were no abnormalities in vital signs. DIAGNOSES: There was no significant cataplexy and less than 2 sleep-onset rapid eye movements (SOREM) in 2 sleep latency tests, with a mean sleep latency of 2.1 and 4.6 minutes. Other sleep deprivation syndromes were excluded from differential diagnosis and finally, a diagnosis of IH was confirmed according to the criteria of the Third Edition of the International Classification of Sleep Disorders. During the course of the disease, attention-deficit/hyperactive disorder (ADHD) and a gaming disorder also diagnosed. INTERVENTIONS: Pharmacological treatment with modafinil was administered for IH and methylphenidate for ADHD. Cognitive behavioral therapy was performed for the gaming disorder. OUTCOMES: The EDS improved, and sleep attacks were no longer observed. The disruption of daily life caused by the gaming disorder was also reduced. LESSONS: Video recordings of sleep attacks are beneficial for identifying the cause of loss of consciousness. Home video recordings may be helpful in the early diagnosis of IH.

Fatigued but not sleepy? An empirical investigation of the differentiation between fatigue and sleepiness in sleep disorder patients in a cross-sectional study.

OBJECTIVE: Sleepiness and fatigue are common complaints among individuals with sleep disorders. The two concepts are often used interchangeably, causing difficulty with differential diagnosis and treatment decisions. The current study investigated sleep disorder patients to determine which factors best differentiated sleepiness from fatigue. METHODS: The study used a subset of participants from a multi-site study (n = 606), using a cross-sectional study design. We selected 60 variables associated with either sleepiness or fatigue, including demographic, mental health, and lifestyle factors, medical history, sleep questionnaires, rest-activity rhythms (actigraphy), polysomnographic (PSG) variables, and sleep diaries. Fatigue was measured with the Fatigue Severity Scale and sleepiness was measured with the Epworth Sleepiness Scale. A Random Forest machine learning approach was utilized for analysis. RESULTS: Participants' average age was 47.5 years (SD 14.0), 54.6% female, and the most common sleep disorder diagnosis was obstructive sleep apnea (67.4%). Sleepiness and fatigue were moderately correlated (r = 0.334). The model for fatigue (explained variance 49.5%) indicated depression was the strongest predictor (relative explained variance 42.7%), followed by insomnia severity (12.3%). The model for sleepiness (explained variance 17.9%), indicated insomnia symptoms was the strongest predictor (relative explained variance 17.6%). A post hoc receiver operating characteristic analysis indicated depression could be used to discriminate fatigue (AUC = 0.856) but not sleepiness (AUC = 0.643). CONCLUSIONS: The moderate correlation between fatigue and sleepiness supports previous literature that the two concepts are overlapping yet distinct. Importantly, depression played a more prominent role in characterizing fatigue than sleepiness, suggesting depression could be used to differentiate the two concepts.

Risk stratification for frailty, impairment and assessment of sleep disorders in community-dwelling older adults.

BACKGROUND: Frailty is associated with an increased susceptibility to functional decline, impairment, hospitalization, and mortality among the older adults. However, the potential reversibility of frailty lies in identifying modifiable factors that could prevent, mitigate, or interrupt its progression. While there is a suggestion that sleep disorders may increase the risk of frailty and impairment, the risk stratification of this relationship remains inconclusive. OBJECTIVE: Stratify the risk of frailty and impairment and investigate potential connections with sleep quality, excessive daytime sleepiness, and the risk of obstructive sleep apnea in older adults dwelling in the community. METHODS: This was a quantitative cross-sectional investigation. Frailty risk and impairment were stratified using the Frail Non-disabled Questionnaire (for impairment) and the FRAIL Scale (for Frailty). The assessment of excessive daytime sleepiness, sleep quality, and the risk of obstructive sleep apnea involved the employment of the Epworth Sleepiness Scale, Pittsburgh Sleep Quality Index, and the STOP-BANG questionnaire, respectively. RESULTS: A total of 109 older adults living in the urban area (86 %, p = 0.010), females (61 %; p = 0.030), median age 68 (64-75) years, with overweight (36 %, p < 0.010) and self-identified as belonging to other racial or ethnic categories (71 %, p < 0.010). According to the impairment assessment, 32 % of participants were classified as disable (p < 0.01). Conversely, as per the frailty evaluation, 33 % were pre-frail and 25 % were identified as frail. Additionally, a substantial proportion experienced poor sleep quality (80 %, p = 0.010), exhibited a moderate risk of obstructive sleep apnea (49 %, p < 0.010), and showed no signs of excessive daytime sleepiness (62 %, p < 0.010). There was a modest correlation between frailty and impairment with poor sleep quality (rho = 0.39; p < 0.001) and the risk of obstructive sleep apnea (rho = 0.26; p = 0.000). However, the was no significant relationship was observed between frailty and impairment and excessive daytime sleepiness (rho = 0.04; p = 0.660). Similarly, a modest correlation was observed between sleep quality (rho = 0.33; p < 0.001), the risk of obstructive sleep apnea (rho = 0.27; p = 0.001), and frailty. Conversely, no correlation was found with excessive daytime sleepiness (rho = 0.05; p = 0.590). Also, the poor sleep quality and the risk of obstructive sleep apnea explain 14 % of the risk of frailty in the population of community-dwelling older adults (r2 = 0.14; p = 0.04). CONCLUSION: This study reveals a modest risk of frailty and impairment with sleep quality and the risk of obstructive sleep apnea, but not with excessive daytime sleepiness in community-dwelling older adults.

Creation of a shortened version of the Sleep Disorders Questionnaire (SDQ).

The 176-item Sleep Disorders Questionnaire (SDQ) was initially developed using canonical discriminant function analysis on 4 groups of sleep disorder patients, but it was never studied by factor analysis in its entirety. Several authors have criticized 2 of its subscales as being confounded with each other, and its narcolepsy scale as substantially over-diagnosing narcolepsy. This study describes its first exploratory factor analysis (EFA), the intent of which was to reassess item membership on the 4 existing subscales and to derive new scales to improve differential diagnosis between patient groups. It was also hoped that EFA could reduce the total number of questions, to increase speed of completion. The EFA was performed on the anonymized SDQ results from a retrospective review of the charts of 2131 persons from 7 sleep disorders clinics and research centers. Factors were assessed via scree plots and eigenvalues. The EFA identified four main factors: insomnia, daytime sleepiness, substance use, and sleep-disordered breathing. The insomnia factor had 3 subfactors: psychological symptoms of insomnia, subjective description of insomnia, and insomnia due to periodic limb movements. The sleepiness factor had two subfactors: daytime sleepiness and neurological symptoms of narcolepsy. The novel substance use factor was homogeneous, as was the sleep-disordered breathing factor. Importantly, the EFA reassigned items from the original SDQ's NAR, PSY, and PLM subscales to five of the new subscales. The Sleep Apnea (SA) subscale emerged mostly unchanged. The 7 resulting factors comprised only 66 items of the original 176-item SDQ. These results have allowed the creation of a new shorter questionnaire, to be called the SDQ-2. External validation of the SDQ-2 is currently underway. It will likely prove to be a superior differential diagnostic instrument for sleep disorders clinics, compared to the original SDQ.

Long sleep time and excessive need for sleep: State of the art and perspectives.

The mechanisms underlying the individual need for sleep are unclear. Sleep duration is indeed influenced by multiple factors, such as genetic background, circadian and homeostatic processes, environmental factors, and sometimes transient disturbances such as infections. In some cases, the need for sleep dramatically and chronically increases, inducing a daily-life disability. This "excessive need for sleep" (ENS) was recently proposed and defined in a European Position Paper as a dimension of the hypersomnolence spectrum, "hypersomnia" being the objectified complaint of ENS. The most severe form of ENS has been described in Idiopathic Hypersomnia, a rare neurological disorder, but this disabling symptom can be also found in other hypersomnolence conditions. Because ENS has been defined recently, it remains a symptom poorly investigated and understood. However, protocols of long-term polysomnography recordings have been reported by expert centers in the last decades and open the way to a better understanding of ENS through a neurophysiological approach. In this narrative review, we will 1) present data related to the physiological and pathological variability of sleep duration and their mechanisms, 2) describe the published long-term polysomnography recording protocols, and 3) describe current neurophysiological tools to study sleep microstructure and discuss perspectives for a better understanding of ENS.

Objective evaluation of excessive daytime sleepiness.

Excessive daytime sleepiness (EDS) is multifactorial. It combines, among other things, an excessive propensity to fall asleep ("physiological sleepiness") and a continuous non-imperative sleepiness (or drowsiness/hypo-arousal) leading to difficulties remaining awake and maintaining sustained attention and vigilance over the long term ("manifest sleepiness"). There is no stand-alone biological measure of EDS. EDS measures can either capture the severity of physiological sleepiness, which corresponds to the propensity to fall asleep, or the severity of manifest sleepiness, which corresponds to behavioral consequences of sleepiness and reduced vigilance. Neuropsychological tests (The psychomotor vigilance task (PVT), Oxford Sleep Resistance Test (OSLeR), Sustained Attention to Response Task (SART)) explore manifest sleepiness through several sustained attention tests but the lack of normative values and standardized protocols make the results difficult to interpret and use in clinical practice. Neurophysiological tests explore the two main aspects of EDS, i.e. the propensity to fall asleep (Multiple sleep latency test, MSLT) and the capacity to remain awake (Maintenance of wakefulness test, MWT). The MSLT and the MWT are widely used in clinical practice. The MSLT is recognized as the "gold standard" test for measuring the severity of the propensity to fall asleep and it is a diagnostic criterion for narcolepsy. The MWT measures the ability to stay awake. The MWT is not a diagnostic test as it is recommended only to evaluate the evolution of EDS and efficacy of EDS treatment. Even if some efforts to standardize the protocols for administration of these tests have been ongoing, MSLT and MWT have numerous limitations: age effect, floor or ceiling effects, binding protocol, no normal or cutoff value (or determined in small samples), and no or low test-retest values in some pathologies. Moreover, the recommended electrophysiological set-up and the determination of sleep onset using the 30­sec epochs scoring rule show some limitations. New, more precise neurophysiological techniques should aim to detect very brief periods of physiological sleepiness and, in the future, the brain local phenomenon of sleepiness likely to underpin drowsiness, which could be called "physiological drowsiness".

Narcolepsy Severity Scale-2 and Idiopathic Hypersomnia Severity Scale to better quantify symptoms severity and consequences in Narcolepsy type 2.

STUDY OBJECTIVES: Narcolepsy type 2 (NT2) is an understudied central disorder of hypersomnolence sharing some similarities with narcolepsy type 1 and idiopathic hypersomnia (IH). We aimed: (1) to assess systematically the symptoms in patients with NT2, with self-reported questionnaires: Epworth Sleepiness Scale (ESS), Narcolepsy Severity Scale (NSS), IH Severity Scale (IHSS), and (2) to evaluate the responsiveness of these scales to treatment. METHODS: One hundred and nine patients with NT2 (31.4 ±â€…12.2 years old, 47 untreated) diagnosed according to ICSD-3 were selected in a Reference Center for Narcolepsy. They all completed the ESS, subgroups completed the modified NSS (NSS-2, without cataplexy items) (n = 95) and IHSS (n = 76). Some patients completed the scales twice (before/during treatment): 42 ESS, 26 NSS-2, and 30 IHSS. RESULTS: Based on NSS-2, all untreated patients had sleepiness, 58% disrupted nocturnal sleep, 40% hallucinations, and 28% sleep paralysis. On IHSS, 76% reported a prolonged nocturnal sleep, and 83% sleep inertia. In the independent sample, ESS and NSS-2 scores were lower in treated patients, with same trend for IHSS scores. After treatment, ESS, NSS-2, and IHSS total scores were lower, with a mean difference of 3.7 ±â€…4.1, 5.3 ±â€…6.7, and 4.1 ±â€…6.2, respectively. The minimum clinically important difference between untreated and treated patients were 2.1 for ESS, 3.3 for NSS-2, and 3.1 for IHSS. After treatment, 61.9% of patients decreased their ESS > 2 points, 61.5% their NSS-2 > 3 points, and 53.3% their IHSS > 3 points. CONCLUSIONS: NSS-2 and IHSS correctly quantified symptoms' severity and consequences in NT2, with good performances to objectify response to medications. These tools are useful for monitoring and optimizing NT2 management, and for use in clinical trials.

Is there a role for repeating the multiple sleep latency test across childhood when initially non-diagnostic?

BACKGROUND: The gold standard investigation for central disorders of hypersomnolence is the Multiple Sleep Latency Test (MSLT). As the clinical features of these disorders of hypersomnolence evolve with time in children, clinicians may consider repeating a previously non-diagnostic MSLT. Currently there are no guidelines available regards the utility and timing of repeating paediatric MSLTs. METHODS: Retrospective review of children aged 3-18years with ≥2MSLTs between 2005 and 2022. Narcolepsy was defined as mean sleep latency (MSL) <8min with ≥2 sleep onset REM (SOREM); idiopathic hypersomnia (IH) was defined as MSL <8min with <2 SOREM. MSLTs not meeting these criteria were labelled non-diagnostic. RESULTS: 19 children (9 female) with initial non-diagnostic MSLT underwent repeat MSLT, with 6 proceeding to a 3rd MSLT following 2 non-diagnostic MSLTs. The 2nd MSLT resulted in diagnosis in 6/19 (32 %) (3 narcolepsy, 3 IH); and 2/6 (33 %) 3rd MSLT were diagnostic (2 IH). Median age at initial MSLT was 7.5y (range 3.4-17.8y), with repeat performed after median of 2.9y (range 0.9-8.2y), and 3rd after a further 1.9 years (range 1.2-4.2y). Mean change in MSL on repeat testing was -2min (range -15.5min to +4.9min, p = 0.18). Of the 8 diagnostic repeat MSLTs, in addition to the MSL falling below 8 min, 2 children also developed ≥2 SOREM that had not been previously present. CONCLUSIONS: A third of repeat MSLTs became diagnostic, suggesting repeat MSLT should be considered in childhood if clinical suspicion persists. Further work needs to address the ideal interval between MSLTs and diagnostic cut-points specific to the paediatric population.

Sleep disorders and mortality: A prospective study in the Canadian longitudinal study on aging.

BACKGROUND AND OBJECTIVES: Sleep disorders are commonly linked to various health conditions, although it remains unclear to what degree they are linked with overall mortality. We compared mortality in different self-reported sleep disorders in a large population-based prospective study. METHODS: In this case-control study within the CLSA cohort, participants completed a questionnaire at baseline (2011-2015) measuring overall sleep satisfaction, daily sleep duration, sleep-onset and sleep-maintenance insomnia, daytime somnolence, REM sleep behavior disorder (RBD), restless leg syndrome (RLS), and obstructive sleep apnea (OSA). The vital status of participants was assessed in July 2019. Baseline sleep problems of participants who died (cases) were compared to those who survived (controls). For each case, five age/sex-matched controls were selected. Binary logistic regression was used to estimate the association between sleep symptoms and mortality, adjusting for age, sex, marital status, province, education, alcohol consumption, smoking, caffeine, and body mass index. In a complementary model, anxiety and depression were also added. RESULTS: Among 30,097 participants at baseline, 974 deaths were reported in 2019 (60.7 % male, age = 72.3 ± 9.4 years). In the initial analysis, mortality cases reported more baseline sleep-maintenance insomnia (12.1 % vs. 8.0 %, Adjusted OR[95%CI] = 1.62[1.15,2.29]), daytime somnolence (2.4 % vs. 1.1 %, AOR = 2.70[1.34,5.44]), and higher possible RLS (16.4 % vs. 12.4 %, AOR = 1.50[1.09,2.05]). They were also more likely to screen positive for possible OSA (33.8 % vs. 24.2 %, AOR = 1.32[1.07,1.64]); however, this effect was not related to core apnea symptoms. Sleep durations exceeding 10 h/day were also associated with increased mortality (3.4 % vs. 1.9 %, AOR = 1.83[1.04,3.24]). Other sleep symptoms/disorders, such as sleep-onset insomnia (7.3 % vs. 4.3 %, AOR = 1.54 [1.00,2.37]), possible RBD (5.3 % vs. 5.1 %, AOR = 1.02[0.62,1.69]), and overall sleep dissatisfaction (26.5 % vs. 22.6 %, AOR = 1.14[0.93,1.41]) were not different among these groups. After adding anxiety and depression to the adjustment model, all differences attenuated to become statistically non-significant, except for daytime somnolence disorder. When stratified by sex, the association between sleep disorders and mortality was only observed in women, with men showing no association. DISCUSSION: We confirm a relationship between numerous sleep disorders and mortality. This effect is most evident in women, and appears to be strongly related to co-existing anxiety and depression.

Sleepy Kids: are the current diagnostic criteria for multiple sleep latency tests enough?

Excessive daytime sleepiness (EDS) is common in childhood and is currently quantified using adult criteria on a multiple sleep latency test (MSLT). This study aimed to describe paediatric MSLT results, particularly focussing on a previously proposed alternative mean sleep latency (MSL) threshold for children of 12 min, and assess the impact of a 5th nap. We performed a retrospective analysis of MSLTs at a single paediatric centre from 2004 to 2021. Narcolepsy was defined as a mean sleep latency (MSL) ≤8min with ≥2 sleep onset REM (SOREM) periods. Idiopathic Hypersomnia (IH) was defined as a MSL ≤8min with <2 SOREMs. An ambiguous MSLT result was defined as a MSL 8-12min and/or ≥2 SOREM periods. Of 214 MSLTs [50 % female, median age 14.0y (range 3.3-20.1y)], narcolepsy was diagnosed in 48 (22 %), IH in 22 (10 %) and the result was ambiguous in 44 (21 %). Those with ambiguous MSLT results were older (15.6 vs 13.4y, p = 0.006) with a higher proportion of females (61 % vs 35 %, p = 0.01) in comparison to the narcolepsy group. A 5th nap was performed in 60 (28 %) of MSLTs and only changed the outcome in one case. In conclusion, MSLT results are borderline in 21 % of paediatric cases, suggesting that current adult diagnostic criteria may miss narcolepsy and IH in children. A 5th nap usually makes no difference or increases the MSL, suggesting that a four nap MSLT protocol could be used apart from rare cases where the result is borderline after the 4th nap.

Poor sleep quality and its associated neurocognitive function in children with obesity with or without obstructive sleep apnea.

OBJECTIVE: To evaluate the associations of OSA severity, snoring symptoms, subjective sleep quality, and daytime sleepiness with executive functioning and behaviors in children with obesity. METHODS: This was a cross-sectional study of children aged 8-18 years with obesity and symptoms suggestive of OSA. All participants underwent an overnight polysomnography and completed a set of questionnaires to assess their sleep-related breathing disordered (SRBD) symptoms [Pediatric Sleep Questionnaire (SRBD-PSQ)], sleep quality [Pittsburgh Sleep Quality Index (PSQI)], executive function [Behavior Rating Inventory of Executive Function (BRIEF)], and inattention and hyperactivity symptoms (Conners-3 Parent Short Form). RESULTS: A total of 85 children (62% male, mean age: 13.9 ± 3.0 years) were included in this analysis, of whom 36, 16, and 33 were categorized into the non-OSA (obstructive apnea hypopnea index, OAHI < 1.5/h), mild OSA (OAHI 1.5-5/h), and moderate-severe OSA (OAHI ≥ 5/h) groups, respectively. Of 85 participants, 27 (32%) were classified with poor sleep quality (PSQI composite score ≥ 8). From multiple linear regression analyses, poor sleep quality and sleepiness were both independently associated with higher BRIEF behavioral regulation T-score, metacognition T-score, and global executive composite T-score in the fully adjusted model. In addition, poor sleep quality was also independently associated with higher Conners-3 inattention and executive functioning T-scores, while greater sleepiness was also associated with a higher learning problem T-score. The presence of OSA and snoring were not associated with any cognitive outcomes. CONCLUSIONS: Subjective sleep quality and daytime sleepiness, but not OSA severity and snoring symptoms, were independently associated with executive functioning and behavioral problems in children with obesity.

Fractional exhaled nitric oxide-a possible biomarker for risk of obstructive sleep apnea in snorers.

STUDY OBJECTIVES: Airway inflammation in patients with obstructive sleep apnea (OSA) has been described and can be assessed by measuring the biomarker fractional exhaled nitric oxide (FeNO). In this pilot study, we investigated FeNO measurements in identification of OSA among persons with snoring. METHODS: In this study we aimed to investigate (1) if FeNO could be used in screening for OSA, (2) if daytime sleepiness correlated to FeNO levels, and (3) whether asthma affected FeNO levels. Persons with snoring were prospectively included in three primary care ear, nose, and throat clinics. Patients underwent spirometry, FeNO tests, and partial polygraphy. They filled out questionnaires on sinonasal and asthma symptoms, daytime sleepiness, and quality of life. Current smokers, patients with upper airway inflammatory conditions, and patients treated with steroids were excluded. RESULTS: Forty-nine individuals were included. Median apnea-hypopnea index was 11.4, mean age was 50.9 years, and 29% were females. OSA was diagnosed in 73% of the patients of whom 53% had moderate-severe disease. Patients with moderate-severe OSA had significantly higher FeNO counts than patients with no or mild OSA (P = .024). Patients younger than 50 years with a FeNO below 15 had the lowest prevalence of moderate-severe OSA. No correlation was found between FeNO measurements and daytime sleepiness, and asthma did not affect FeNO levels. CONCLUSIONS: We found a low prevalence of moderate-severe OSA in persons with snoring when FeNO and age were low. This might be considered in a future screening model, though further studies testing the FeNO cutoff level and the diagnostic accuracy are warranted. CLINICAL TRIAL REGISTRATION: Registry: ClinicalTrials.gov; Name: NO Measurements in Screening for Asthma and OSA, in Patients With Severe Snoring; URL: https://clinicaltrials.gov/study/NCT03964324; Identifier: NCT03964324. CITATION: Kiaer E, Ravn A, Jennum P, et al. Fractional exhaled nitric oxide-a possible biomarker for risk of obstructive sleep apnea in snorers. J Clin Sleep Med. 2024;20(1):85-92.

Excessive daytime sleepiness in young and middle-aged Chinese adults with obstructive sleep apnea: implications for cognitive dysfunction.

OBJECTIVE: The objective of this study was to investigate the effects of excessive daytime sleepiness (EDS) on cognitive function among Chinese young and middle-aged Chinese patients with obstructive sleep apnea (OSA). METHODS: Chinese adults struggling from moderate to severe OSA with apnea-hypopnea index (AHI) ≥ 15 events per hour and adults with primary snoring and mild OSA (AHI < 15 events per hour) were included in the study. The Epworth Sleepiness Scale measured hypersomnia, and cognitive function was assessed using the Mini-mental State Examination (MMSE) and Montreal Cognitive Assessment (MOCA). RESULTS: In comparison to the primary snoring and mild OSA group (n=635), the moderate to severe OSA group (n=1423) tended to be older men with higher scores on the Epworth Sleepiness Scale (ESS), as well as higher levels of oxygen desaturation (ODI) and a higher body mass index (BMI). Patients with moderate to severe OSA had fewer years of education, lower minimum arterial oxygen saturation (min-SaO2), and more severe sleep disturbances, such as decreased slow wave sleep (SWS) and rapid eye movement (REM) and increased non-REM stages (N1 and N2). Comorbid conditions such as hypertension and diabetes mellitus were more common in these patients (P < 0.01 and P < 0.05, accordingly). Only the delayed recall scores were statistically lower in the moderate to severe OSA group than the primary snoring and mild OSA group (P < 0.05). The main factor associated with delayed recall was the ESS score rather than age or years of education among moderate-severe OSA patients ≤ 40 years of age (P < 0.05). After controlling for potential confounding factors such as age, gender, BMI, education, hypertension, diabetes, sleep stages (SWS and REM), minimum arterial oxygen saturation (min-SaO2), oxygen ODI, and AHI, there was a negative correlation between the Epworth Sleepiness Scale (ESS) score and the delayed recall scores. CONCLUSION: Patients with moderate to severe OSA had cognitive dysfunction, particularly impairment of delayed recall. Excessive daytime sleepiness (EDS) was significantly associated with cognitive dysfunction in young and middle-aged patients with OSA.