Sleep positions in children with Down syndrome and obstructive sleep apnea

ObjectivesTo assess sleep positions in children with both Down syndrome (DS) and obstructive sleep apnea (OSA) and determine if there is a preferred sleep position by severity of apnea.MethodsA single-center retrospective review of patients with both DS and OSA was performed. Caregivers reported sleep position utilized greater than 50% of observed sleep time. Accuracy of this report was confirmed through review of hypnograms from polysomnography studies.ResultsEighty-two patients met inclusion criteria. Median body mass index (BMI) was 26.6 and 56% of patients had a prior tonsillectomy and/or adenoidectomy. The mean obstructive AHI (OAHI) was 25.33 with 90.4% having severe OSA, 9.6% having moderate OSA, and no patients having mild OSA. Reported sleep positions were skewed towards lateral/decubitus (82.9%) compared to prone (11.0%) and supine (6.1%). This was consistent with hypnogram data where 71% of total sleep time in lateral/decubitus positions compared to prone (13%) and supine (6%). The median changes in sleep position per patient was 5 (IQR: 3–6). Lower BMI (p < 0.001, 95% CI: 0.32–1.13) and tonsillectomy (p < 0.001, 95% CI: 7.7–18.19) were associated with lower OAHI. Sleep position was not associated with age (p = 0.19), sex (p = 0.66), race (p = 0.10), ethnicity (p = 0.68) nor history of tonsillectomy (p = 0.34). Preferred sleep position was not correlated with OAHI (p = 0.78, r = 0.03) or OSA severity (p = 0.72, r = 0.03).ConclusionsThis study highlights the possibility that children with DS may have preferential sleep positions that cater to optimized airflow in the context of OSA although further prospective study is needed.     

Could non-HDL-cholesterol be a better marker of atherogenic dyslipidemia in obstructive sleep apnea?

Background/objectiveObstructive sleep apnea (OSA) is independently associated with dyslipidemia, a surrogate marker of atherosclerosis. Low-density lipoprotein (LDL)-cholesterol is accepted as a major independent risk factor for cardiovascular disease. However, non-high-density lipoprotein (HDL)-cholesterol is a better marker of atherogenic dyslipidemia and recommended as a target of lipid lowering therapy. We aimed to assess the prevalence of atherogenic dyslipidemia, and relationship between OSA severity and serum LDL-cholesterol and non-HDL cholesterol levels in OSA patients.MethodsWe retrospectively evaluated treatment naïve 2361 subjects admitted to the sleep laboratory of a university hospital for polysomnography. All subjects’ lipid profile including total cholesterol, LDL-cholesterol, HDL-cholesterol, triglycerides, and non-HDL-cholesterol were measured.ResultsOut of 2361 patients (mean age 49.6 ± 11.9 years; 68.9% male, apnea-hypopnea index 36.6 ± 28.4/h), 185 (7.8%) had no OSA and 2176 (92.2%) had OSA. Atherogenic dyslipidemia prevalence was high (57–66%) in OSA patients, and especially increased in severe OSA compared to other groups (p < 0.05). Though total and LDL-cholesterol did not differ between those with and without OSA, non-HDL-cholesterol (p = 0.020), and triglycerides (p = 0.001) were higher and HDL-cholesterol levels (p = 0.018) were lower in OSA patients than non-OSA. Non-HDL-cholesterol was significantly correlated with OSA severity (p < 0.001) and hypoxia parameters (p < 0.01), whereas LDL-cholesterol showed no correlation.ConclusionsAtherogenic dyslipidemia is highly prevalent and non-HDL-cholesterol levels are significantly increased, predominantly in severe OSA patients. Non-HDL-cholesterol but not LDL-cholesterol, is significantly correlated with OSA severity and hypoxia parameters. Therefore, it could be better to use non-HDL-cholesterol, which is a guideline recommended target of lipid therapy, as a marker of atherosclerotic cardiovascular risk in OSA patients.     

Positional device therapy for the treatment of positional obstructive sleep apnea in children: a pilot study

BackgroundThere is a critical gap in identifying effective interventions for children with obstructive sleep apnea (OSA) who do not tolerate continuous positive airway pressure therapy. Positional OSA (POSA) is a common clinical phenotype whereby OSA occurs predominantly while sleeping in supine position. POSA may be amenable to treatment with a positional device, a belt worn around the chest with cushions on the back to prevent supine positioning, but no data exists in children. The primary aim of this study was to evaluate the efficacy of positional device therapy for the treatment of POSA in children.MethodsThis observational study included children aged 4–18 years with POSA and an obstructive apnea-hypopnea index (OAHI) ≥ 5 events/hour on baseline polysomnogram (PSG) who underwent a second PSG to evaluate the efficacy of a positional device. The primary outcome was the change in OAHI.ResultsTen children were included (8 male, median age 11.2 years, median body mass index z-score 1.6). Compared to the baseline PSG, PSG data obtained while using a positional device showed a reduced median (interquartile range) OAHI (15.2 [8.3–25.6] versus 6.7 [1.0–13.7] events/hour respectively; p = 0.004) and percentage of total sleep time in supine position (54.4 [35.0–80.6]% versus 4.2 [1.1–25.2]% respectively; p = 0.04). Despite observed improvements in the oxygen desaturation index, these results were not statistically significant.Significance and conclusionsIn this novel pilot study, positional device therapy was effective for the treatment of POSA. Positional device therapy may potentially change clinical practice as a cost-efficient and non-invasive treatment option for POSA.     

The impact of obstructive sleep apnea on bronchiolitis severity in children with Down syndrome

ObjectivesAcute bronchiolitis commonly causes respiratory failure in children ≤2 years, and is particularly severe in those with Down syndrome (DS). Obstructive sleep apnea (OSA), common in DS, is also associated with respiratory complications. However, it is unknown whether OSA is associated with worse outcomes in children with and without DS, hospitalized with bronchiolitis. We hypothesized that in children with bronchiolitis, OSA is associated with worse outcomes in those with DS, independent of DS-related comorbidities.MethodsHospital discharge records of children with bronchiolitis aged ≤2 years were obtained for 1997–2012 from the Kid's Inpatient Database. The primary outcome was invasive mechanical ventilation (IMV), and secondary outcomes were non-invasive mechanical ventilation (NIMV), length of hospital stay, and inflation-adjusted cost of hospitalization (IACH). Multivariable regression was conducted to ascertain the associations between OSA and primary and secondary outcomes accounting for DS-associated comorbidities.ResultsThere were 928,961 hospitalizations for bronchiolitis. The DS group with bronchiolitis (n = 8697) was more likely to have OSA [241 (2.77%) vs 1293 (0.14%), p < 0.001] compared to the non-DS group (n = 920,264). Multivariable logistic regression showed that OSA was associated with IMV (adjusted odds ratio [OR], 3.32 [95% CI 2.54–4.35], p < 0.0001) in all children with bronchiolitis; and in those with DS, it was associated with IMV (adjusted OR, 2.34 [95% CI 1.38–3.97], p = 0.002), NIMV (adjusted OR, 8.21 [95% CI 4.48–15.04], p < 0.0001) and IACH (adjusted β, 0.18 [95% CI 0.02–0.34], p = 0.031).ConclusionsOSA is independently associated with assisted ventilation in all children hospitalized with bronchiolitis, regardless of DS-associated comorbidities in those with DS. The severity of bronchiolitis in children with DS may be driven by the high prevalence of OSA.     

Predictors of CPAP adherence following stroke and transient ischemic attack

ObjectiveContinuous positive airway pressure (CPAP) has been shown to improve functional, motor and cognitive outcomes in post-stroke obstructive sleep apnea (OSA). However, rates of CPAP adherence are often low and factors impacting CPAP adherence remain under-explored. Our objective was to determine predictors of CPAP adherence in patients who had a stroke or transient ischemic attack (TIA).MethodsWe screened 313 stroke/TIA patients for OSA using in-hospital polysomnography or the ApneaLink home sleep apnea test. Potential predictors were recorded at baseline and adherence to CPAP was recorded during a six-month follow-up visit. Selected variables from our univariate analyses were included in multivariate regression models to determine predictors of CPAP adherence. For our logistic regression analyses, CPAP adherence (CPAP use of ≥4 h per night) was the dependent outcome variable. In our linear regression analyses, total CPAP use per week (recorded in hours) was the dependent outcome variable.ResultsEighty-eight patients (mean age 67.81 ± 13.09 years, 69.32% male, mean body mass index 27.93 ± 5.23 kg/m²) were diagnosed with OSA, prescribed CPAP, and assessed for adherence at a six-month follow-up visit. In these 88 patients, 46 (52.27%) were adherent with CPAP therapy. From our regression models, two significant predictors of CPAP adherence were identified: greater functional status (p = 0.04) and not endorsing daytime tiredness (p = 0.047) post-stroke/TIA.ConclusionPatients with greater functional capacity and those with less daytime fatigue demonstrated stronger adherence to CPAP therapy. Our findings may facilitate future treatment strategies for enhancing CPAP adherence in the vulnerable stroke/TIA population.     

Wake-up stroke is not associated with obstructive sleep apnea

Objective/BackgroundObstructive sleep apnea is a risk factor for stroke. This study sought to assess the relationship between obstructive sleep apnea (OSA) and wake-up strokes (WUS), that is, stroke symptoms that are first noted upon awakening from sleep.Patients/methodsIn this analysis, 837 Brain Attack Surveillance in Corpus Christi (BASIC) project participants completed an interview to ascertain stroke onset during sleep (WUS) versus wakefulness (non-wake-up stroke, non-WUS). A subset of 316 participants underwent a home sleep apnea test (HSAT) shortly after ischemic stroke to assess for OSA. Regression models were used to test the association between OSA and WUS, stratified by sex.ResultsOf 837 participants who completed the interview, 251 (30%) reported WUS. Among participants who underwent an HSAT, there was no significant difference in OSA severity [respiratory event index (REI)] among participants with WUS [median REI 17, interquartile range (IQR) 10, 29] versus non-WUS (median REI 18, IQR 9, 30; p = 0.73). OSA severity was not associated with increased odds of WUS among men [unadjusted odds ratio (OR) 1.011, 95% confidence interval (95% CI) 0.995, 1.027] or women (unadjusted OR 0.987, 95% CI 0.959, 1.015). These results remained unchanged after adjustment for age, congestive heart failure, body mass index, and pre-stroke depression in men (adjusted OR 1.011, 95% CI 0.994, 1.028) and women (adjusted OR 0.988, 95% CI 0.959, 1.018).ConclusionsAlthough OSA is a risk factor for stroke, the onset of stroke during sleep is not associated with OSA in this large, population-based stroke cohort.     

The relationship between gross motor function impairment in cerebral palsy and sleeping issues of children and caregivers

AimTo investigate, among children and adolescents with cerebral palsy (CP), the relationship between impairment of the gross motor function and: (i) child sleep disorders; (ii) the need for nocturnal support; and (iii) the quality of sleep of their caregivers.MethodsFor children, we considered their scores on the gross motor function measure (GMFM-88) and on the sleep disturbance scale for children (SDSC), besides analyzing qualitative features about their sleep. For caregivers, we considered their scores in the Pittsburgh sleep quality index (PSQI).ResultsOur sample was comprised of 87 participants with mean age of 11.4 years old (±3.4). We observed correlations between GMFM-88 and disorders of initiating and maintaining sleep (DIMS) (r = −0.22; p = 0.039), sleep–wake transition disorders (SWTD) (r = 0.26; p = 0.017) and disorders of arousal (DA) (r = 0.23; p = 0.033). Children receiving nocturnal support presented lower scores in the GMFM-88 (p = 0.001) and higher scores in the SDSC (p = 0.029). For the caregivers, we found no correlation between GMFM-88 and PSQI. Nonetheless, their PSQI scores correlated with the SDSC scores (r = 0.24; p = 0.027).ConclusionImpairment of the gross motor function correlated with DIMS and the need for nocturnal support but might not have an impact on the caregivers’ sleep, which in turn correlated with child sleep disorders.     

Prevention of leakage due to mouth opening through applying an oral shield device (Sominpax™) during nasal CPAP therapy of patients with obstructive sleep apnea

Background/objectiveThe first line treatment for obstructive sleep apnea (OSA) is nasal continuous positive airway pressure (nCPAP), for which a variety of masks are available. While nasal masks (NM) are the first choice; oronasal masks (ONM) are also frequently used to prevent mouth dryness resulting from mouth opening. Our cross-sectional, prospective, randomized, un-blinded study addressed the efficacy of wearing an oral shield in addition to NM in preventing mouth leakageMethodsPatients with OSA and established therapy using NM and complaining about mouth dryness (n = 29) underwent three polysomnographies (PSGs) using NM, ONM or a nose mask in combination with an oral shield (NMS). Mask leakage was continuously documented and objective sleep quality was assessed.ResultsThere were significant differences in the apnea-hypopnea-index (AHI) between ONM (8.5/h; SD 6,7) and NM/nasal mask combined with oral shield device (NMS) (2.6/h; SD 2,3; 2.7/h; SD 2,6) (p < 0,05) as well as in leakage [ONM (39.7 l/min SD 12,4); NM (34.6 l/min SD 9,4); NMS (33.1 l/min SD 9,6)] (p = 0.011). Furthermore, analysis of sleep quality (NREM3) favored NM and NMS over ONM (p = 0.02). There were no significant differences between NM and NMS in any objective outcome.ConclusionsOur data consistently confirmed the NM as the first choice for continuous positive airway pressure (CPAP) therapy of OSA. Notably, we demonstrated a high potential of the oral shield for patients with mouth opening to achieve additional comfort and thereby possibly compliance, without affecting nCPAP therapy effectiveness.     

Inflammation in children with neuromuscular disorders and sleep disordered breathing

IntroductionPaediatric obstructive sleep apnoea is associated with systemic inflammation and co-morbidities. We assessed whether sleep disordered breathing (SDB) due to neuromuscular weakness was associated with elevated airway and systemic pro-inflammatory cytokines.MethodsConsecutive neuromuscular children (age 5–18years) underwent overnight full polysomnography and morning collection of serum and breath condensate, analysed for cytokines (Interleukin-10, Interleukin-6, Interleukin-1β, Tumour Necrosis Factorα, high-sensitivity C-Reactive Protein, Intercellular and Vascular Adhesion Molecules ICAM-1, VCAM-1). Cytokine levels were related to Oxygen desaturation index (ODI), desaturation>4%/h, and levels of transcutaneous carbon dioxide overnight (tcCO2≥6.7 kPa > 2% sleep).ResultsA total of 23 patients were included, median age 12.6 years (IQR 8.7–14.6). ODI>3/h was associated with higher breath and serum IL-6 (p = 0.02). Children with elevated CO₂ overnight had higher ICAM-1 and VCAM-1. CO₂ levels correlated with serum ICAM-1 (rs0.570, p = 0.026) and VCAM-1 (rs0.76, p = 0.001).DiscussionSDB in neuromuscular children is associated with raised serum IL-6, VCAM-1, ICAM-1. This may predispose these children to future cardiovascular and other co-morbidities.     

Patterns of nightly CPAP usage in OSA patients with suboptimal treatment adherence

Background and objectivesLow adherence impairs the effectiveness of continuous positive airway pressure (CPAP) therapy in patients with obstructive sleep apnoea (OSA), but knowledge on CPAP usage micro-patterns is mostly lacking. Thus, the aim of this study was to analyse usage micro-patterns among patients with suboptimal CPAP adherence.MethodsWe analysed CPAP usage datasets comprising the initial 31 nights of therapy. By employing a threshold of 4 h usage in at least 70% of nights, we subdivided the patients into suboptimal and optimal users. We investigated single CPAP start- and stop-points, and introduced the parameter “interruption-rate”, by dividing the amount of therapy interruptions per night by the usage duration per night. This parameter represents the amount of interruptions per 1 h of CPAP usage. Group comparison analysis was performed via t-test, Wilcoxon rank sum-test, and via Chi²-test.ResultsWe included datasets of 48 suboptimal and 48 optimal users (55.9 ± 11.3 years, 83.3% men) in the analysis. Interruption-rate was significantly higher among suboptimal users, when compared with optimal users (median (quartiles) 0.24 (0.14/0.45) versus 0.15 (0.05/0.28), p < 0.0011). Suboptimal users were more likely to report that CPAP reduced their sleep quality, waked them up at night, and that CPAP side effects or problems with the device impaired their adherence.ConclusionsCPAP usage micro-patterns are more fragmented among OSA patients with lower overall adherence. These patterns might result from impaired sleep quality, due to CPAP side effects, and device-associated problems.     

Does neck circumference predict obstructive sleep apnea in children with obesity?

ObjectivesObstructive sleep apnea (OSA) affects 10–50% of children with obesity, but its identification is challenging and wait times for testing are long. Previous studies suggest that neck circumference (NC) and neck-to-height ratio (NHR) may predict OSA. Our objectives were to 1) evaluate associations of NC and NHR with OSA; 2) model NHR as a predictor of OSA, adjusting for age, sex, and Tanner stage; and 3) identify thresholds of NHR associated with OSA, in children with obesity.MethodsParticipants were aged 8–17 years, with obesity (BMI >95%ile), undergoing polysomnography. Associations between NC and NHR with OSA were evaluated. NHR, age, sex and self-reported Tanner stage (early/late) were included in a negative binomial multiple regression model to predict obstructive apnea hypopnea index (OAHI).Results71 children participated, with median age 14.8 years (IQR 12.6, 16.0), 54% male, median BMI z-score 2.5 (IQR 2.3, 2.7), and 77% late Tanner stage. OSA was severe in 18 children (25.4%), moderate in 12 (16.9%), and mild in 18 (25.4%). In the model, each 0.01 increase in NHR was associated with a 55% increase in OAHI (95% CI: 36%, 80%); boys had a 119% higher OAHI than girls (95% CI: 10%, 337%). Threshold NHR associated with moderate-severe OSA was 0.25 in females and 0.23 in males.ConclusionsNHR and male sex independently predict OSA severity in children with obesity, adjusting for age and Tanner stage. Children with obesity and NHR above identified thresholds are more likely to have moderate-severe OSA, and may benefit from earlier polysomnography.     

Diagnostic approach to sleep disordered-breathing among patients with grade III obesity

Sleep apnea test (SAT) is a cost-effective approach to evaluate subjects without associated comorbidities suspected for obstructive sleep apnea (OSA), a disorder particularly common in obese subjects. The association of obesity with awake hypercapnia (carbon dioxide arterial pressure, PaCO₂ ≥45 mmHg) defines the obesity-hypoventilation syndrome (OHS), which in turn results in increased morbidity and mortality compared to simple OSA. Isolated hypoventilation during sleep in obese patients (obesity-related sleep hypoventilation, ORSH) is now considered as an early stage of OHS. The aim of this study was to assess the performance of SAT in diagnosing OSA and predicting the presence of ORHS among patients with grade III obesity without awake hypercapnia.MethodsOver a 14-months period, patients with grade III obesity (body mass index≥40 kg/m²) presenting moderate-to-severe OSA (apnea-hypopnea index [AHI]≥15) upon SAT and normal awake PaCO₂ at arterial blood gas analysis, systematically underwent in-lab nocturnal polysomnography combined with transcutaneous carbon dioxide pressure (PtcCO₂) monitoring.ResultsAmong 48 patients included in the study, 16 (33%) presented an AHI<15 upon polysomnography and 14 (29%) had ORSH. The test revealed no difference in ORSH prevalence between patients with AHI <15 or ≥15 (31% vs. 25%). No SAT variables were independently associated with increased PtCO₂.ConclusionsThis study shows that SAT overestimates OSA severity and ORSH affects one third of patients with grade III obesity without awake hypercapnia and with moderate-to-severe OSA at SAT, suggesting how polysomnography combined with PtCO₂ monitoring is the most appropriate diagnostic approach for OSA and ORSH in this population.     

Association of visceral adiposity and systemic inflammation with sleep disordered breathing in normal weight,never obese adolescents

Objective/backgroundWhile obesity is a known risk factor for sleep disordered breathing (SDB), a large proportion of children with SDB are not overweight as per body mass index percentile (BMI%) criteria. This study aimed to examine whether premorbid or concurrent adiposity phenotypes and inflammation are associated with SDB in normal weight youth.Patients/methodsA total of 242 persistently non-overweight (BMI%<85) subjects from the Penn State Child Cohort (PSCC, N = 421, 5-12 y at baseline and 12-23 y at follow-up), were studied. The apnea/hypopnea index (AHI) was ascertained via polysomnography (PSG) at both time points. At follow-up, a dual-energy X-ray absorptiometry (DXA) scan assessed android and gynoid distribution and subcutaneous (SAT) and visceral (VAT) adiposity composition, while a fasting blood draw was assayed for C-reactive protein (CRP) and interleukin-6 (IL-6) levels. Multivariable linear regression models with AHI at follow-up as primary outcome were adjusted for sex, race, adenotonsillectomy, age and AHI at baseline.Results and conclusionsIncreased waist circumference (β = 0.227, p = 0.001) at baseline, but not BMI%, neck or hip circumference, was significantly associated with a higher AHI at follow-up. VAT (β = 0.309, p < 0.001), IL-6 (β = 0.243, p < 0.001), SAT (β = 0.235, p = 0.013), CRP (β = 0.221, p = 0.001), and an android distribution (β = 0.196, p = 0.003) at follow-up were significantly associated with a higher AHI at follow-up. Childhood central adiposity predicts SDB in adolescence, even in individuals who have never been overweight since childhood as per BMI criteria. Visceral adiposity and inflammation are concurrent to adolescent SDB, which supports the clinical utility of these biomarkers in predicting its associated cardiometabolic risk.     

Long-term prophylactic efficacy of transcranial direct current stimulation in chronic migraine. A randomised,patient-assessor blinded,sham-controlled trial

ObjectiveTo assess the prophylactic effect of anodal tDCS of the left motor cortex in patients with resistant chronic migraine (CM) and its long-term maintenance.MethodsIn a patient-assessor blinded, sham-controlled trial, 36 patients were randomized to receive anodal tDCS (active group, n = 18) or sham tDCS (sham group, n = 18). The studied population was characterized by a previous failure of at least 3 classes of preventive drugs and a mean duration of migraine history of 26 years. The tDCS procedure consisted of an induction phase of 5 consecutive daily sessions (week 1) followed by a maintenance phase of 1 weekly session during the next 4 weeks and two bimonthly sessions in the next month, for a total of 11 sessions during 2 months. Anodal tDCS was delivered at 2 mA intensity for 20 min over the left motor cortex. The primary endpoint was the reduction in the monthly number of migraine attacks from baseline to each period of follow-up (months 1, 2, 3, 5) between the active and sham groups.ResultsThe monthly number of migraine attacks expressed as the percentage of reduction from baseline was significantly reduced in the active versus the sham group, from the end of first month (?21% ± 22 vs. ?2% ±25, p = 0.019) to the end of follow-up (3-month post-treatment) (?32% ± 33 vs. ?6% ±39, p = 0.011). At this time, the rate of responders, defined as a reduction of the monthly number of migraine attacks ≥30% from baseline, was significantly higher in the active group than in the sham group (50% vs. 14%, p = 0.043).ConclusionOur results show a marked prophylactic effect of anodal tDCS of the left motor cortex in resistant CM extending several months after the stimulation period, and suggest that this neuromodulatory approach may be part of the prophylactic alternatives available for CM.     

Maternal obstructive sleep apnea and neonatal birth outcomes in a population based sample

ObjectiveEvaluate the association of OSA with birth outcomes including the risk of congenital anomalies and the need for a higher level of clinical care at delivery.MethodsPopulation-based study that linked newborn records with maternal records. Data from 95 perinatal centers across all geographic census divisions of the U.S. of women with a delivery diagnosis from 2010 to 2014 whose records could be linked to the corresponding newborn record. An International Classification of Diseases, ninth Revision (ICD-9) code for sleep apnea was used to identify exposure and outcome variables. Univariate and multivariate logistic regression analyses were performed with a model that included substance use, obesity, diabetes, maternal co-morbidities, and pregnancy complications.ResultsIn this study, 1,423,099 maternal records were linked to live newborn records. OSA was associated with a higher risk for congenital anomalies in offspring (aOR 1.26, 1.11 to 1.43), with the highest risk being that of musculoskeletal anomalies (aOR 1.89, 1.16 to 3.07) after adjusting for comorbidities and potential teratogens. Neonates born to mothers with OSA were more likely to be admitted to the intensive care unit (25.3% vs. 8.1%, p < 0.001), require resuscitation (aOR 2.76, 1.35 to 5.64) and have a longer hospital stay (aOR 2.25, 1.85 to 2.65).ConclusionsAlthough our study does not establish causation, it is the first to demonstrate a higher risk of congenital anomalies and resuscitation at birth in neonates of mothers with OSA, emphasizing the importance of identifying OSA in pregnant women and women of reproductive age.     

Brain tumours result in sleep disorders in children and adolescents

Background and objectivesSleep disturbances are frequently reported in children with brain tumours. The objective of our cross-sectional study was to systematically examine sleep in these children. We hypothesised that children with tumours involving the sleep-wake-regulatory areas have an altered sleep-wake-regulation.MethodsSixty-one patients aged 0–18 years and with a diagnosis of a primary brain or cervical medullary tumour were included. They were categorised based upon tumour location into two groups – those affecting the sleep-wake regulatory regions, i.e. brain stem, basal forebrain, hypothalamus, thalamus, and posterior fossa compressing the brain stem and those that did not. Sleep history, questionnaire surveys, polysomnography, and multiple sleep latency test were used, as indicated clinically. Surveys included Pediatric Daytime Sleepiness Scale, Children's Sleep Habits Questionnaire, Strengths and Difficulties Questionnaire, and Pediatric Quality of Life Inventory, Multidimensional Fatigue Scale and Generic Core Scale.ResultsPatients with tumours involving the sleep-wake regulatory areas were sleepier/more fatigued (p = 0.03). Sleep apnoea was observed in 86% of all the patients and comorbid narcolepsy in 8%, without group differences (p ≥ 0.12). Patients with tumours involving the sleep-wake-regulatory areas had more emotional problems (p = 0.04), were more affected by mental health problems (p < 0.001), and had poorer quality of life (p ≤ 0.03).ConclusionsMany children with brain tumours suffer from disturbed sleep, poor mental health, and low quality of life. We recommend that systematic sleep evaluation is included in their routine care along with psychological and social support.     

Scalp EEG interictal high frequency oscillations as an objective biomarker of infantile spasms

ObjectiveTo investigate the diagnostic utility of high frequency oscillations (HFOs) via scalp electroencephalogram (EEG) in infantile spasms.MethodsWe retrospectively analyzed interictal slow-wave sleep EEGs sampled at 2,000 Hz recorded from 30 consecutive patients who were suspected of having infantile spasms. We measured the rate of HFOs (80–500 Hz) and the strength of the cross-frequency coupling between HFOs and slow-wave activity (SWA) at 3–4 Hz and 0.5–1 Hz as quantified with modulation indices (MIs).ResultsTwenty-three patients (77%) exhibited active spasms during the overnight EEG recording. Although the HFOs were detected in all children, increased HFO rate and MIs correlated with the presence of active spasms (p < 0.001 by HFO rate; p < 0.01 by MIs at 3–4 Hz; p = 0.02 by MIs at 0.5–1 Hz). The presence of active spasms was predicted by the logistic regression models incorporating HFO-related metrics (AUC: 0.80–0.98) better than that incorporating hypsarrhythmia (AUC: 0.61). The predictive performance of the best model remained favorable (87.5% accuracy) after a cross-validation procedure.ConclusionsIncreased rate of HFOs and coupling between HFOs and SWA are associated with active epileptic spasms.SignificanceScalp-recorded HFOs may serve as an objective EEG biomarker for active epileptic spasms.     

Bidirectional association between alopecia areata and sleep disorders: a population-based cohort study in Taiwan

BackgroundThe relationship between alopecia areata (AA) and sleep disorders remains uncertain. This study aims to investigate the bidirectional association between AA and sleep disorders.MethodsTo assess the risk of developing sleep disorders, we recruited 5648 patients with AA and 22,592 matched controls from the National Health Insurance Research Database (NHIRD) in Taiwan. Similarly, risk of developing AA was assessed in 93,130 patients with sleep disorders (including 7310 patients with obstructive sleep apnea [OSA] and 85,820 patients with non-apnea insomnia) and 372,520 matched controls. Cox regression model was used for the analysis.ResultsAA patients had a significantly increased risk of developing OSA (adjusted hazard ratio [aHR] 3.80; 95% confidence interval [CI] 2.53–5.71) and non-apnea insomnia (aHR 4.20; 95% CI 3.68–4.79). Conversely, presence of sleep disorders significantly increased the risk of AA development (aHR 4.70; 95% CI 3.99–5.54). Both OSA (aHR 3.89; 95% CI 2.46–6.16) and nonapnea insomnia (aHR 4.77; 95% CI 4.03–5.64) were associated an increased risk of developing AA.ConclusionsPatients with AA have a higher risk of developing sleep disorders compared to controls, and vice versa. Further studies are needed to investigate the shared pathogenic mechanism underlying these two conditions.     

Mandibular advancement device in Parkinson's disease: a pilot study on efficacy and usability

Background/objectivesContinuous positive airway pressure (CPAP) is efficacious in the treatment of obstructive sleep apnea syndrome (OSAS) in patients with Parkinson's disease (PD). However, this treatment is often not well tolerated in this disabled population. We explored, in a pilot study, the efficacy, observance, and usability of mandibular advancement device (MAD) for the treatment of OSAS in this peculiar population.Patients/methodsTwenty patients with PD and moderate or severe OSAS were included in the study. Ten patients had refused or not tolerated the validated treatment with CPAP so that they were treated with MAD. The patients treated with MAD were matched for sex, age and body mass index (BMI) to 10 patients with PD treated with CPAP.We explored the efficacy of MAD on sleep disorders complaints (PDSS-2) and on sleep recordings. We compared adherence, tolerance and usability with MAD and with CPAP.ResultsMAD improved sleep complaints increasing PDSS-2 scores (85 [55–106] vs 106 [88–126], p < 0.005), and sleep respiratory measures reducing apnea/hypopnea index (AHI) (50.8 [30.0–76.4] vs 9.4 [5.0–45.2], <0.001) and oxygen desaturation index (22.9 [2.1–92.0] vs 3.8 [0.2–34.2], p < 0.05). Observance was higher with MAD than with CPAP. Usability and caregiver satisfaction were higher with MAD than with CPAP whereas side effects were similarly reported.ConclusionMandibular advancement device may be an noteworthy alternative treatment of OSAS in patients with PD.     

Core body temperature changes in school-age children with circadian rhythm sleep–wake disorder

Objective/backgroundCore body temperature (CBT) is considered a valuable marker for circadian rhythm. This study aimed to investigate the changes in CBT that are associated with the symptoms of circadian rhythm sleep–wake disorder (CRSWD) post-treatment in children.Patients/methodsTwenty-eight school-age children [10 boys and 18 girls; mean age (±standard deviation), 13.68 ± 0.93 years] who were admitted to our hospital with CRSWD underwent treatment for 6–8 weeks according to the following protocol: lights-out for sleep at 21:00; phototherapy for waking at 6:00 or 7:00; light exercise everyday (eg, a 20- to 30-min walk). CBT was continuously measured for 24 h on the first day of admission and on the first day after treatment.ResultsThe mean time of sleep onset/offset (±standard deviation; in hours:minutes) 1 week before admission and 1 week after treatment were 23:53 ± 2:26/9:58 ± 2:15 and 21:17 ± 0:19/6:46 ± 0:32, respectively. The mean times of sleep onset and offset measured post-treatment were significantly earlier than those measured pre-treatment (p < 0.001). The mean CBT and mean minimum CBT during sleep were significantly lower on the first day post-treatment than on the first day of admission (p = 0.011 and p < 0.001, respectively).ConclusionsSymptom improvements in patients with CRSWD were associated with a decrease in CBT during sleep, suggesting that CBT may be a biomarker for improvements in CRSWD. These results help elucidate the cause of this sleep disorder.