Effectiveness of an intervention program on physical activity in children with narcolepsy type 1.

OBJECTIVES: Physical activity (PA) is recommended as part of the management of narcolepsy type 1 (NT1). This study aimed at 1) characterizing PA in children and adolescents treated for NT1 using objective and subjective measurements, 2) evaluating how PA is associated with NT1 symptoms and comorbidities, and 3) evaluating the effects of an Adapted Physical Activity (APA) program on PA and clinical characteristics. PATIENTS/METHODS: Patients with NT1 from the National Reference Center of Narcolepsy (Lyon, France) were consecutively included in an APA intervention protocol. Narcolepsy symptoms and comorbidities were collected using standardized questionnaires and sustained attention was evaluated using the Bron-Lyon Attention Stability Test before and after the four-week APA intervention. PA was measured objectively using actigraphy throughout the study. RESULTS: Twenty-seven NT1 patients were included (median age 14.7 years [8.3-18.4], cataplexy 88.9%, obesity 37.0%). At baseline, 52.4% of the patients had satisfactory PA levels according to international recommendations. Patients with leisure-time PA (LTPA) showed higher quality of life than patients without. 45% of the patients increased PA during the intervention compared to baseline. These responsive patients had more depressive feelings and tended to have lower objective PA than non-responsive patients at baseline. No significant correlation was found between PA levels before and during the intervention and other clinical data. CONCLUSIONS: Most children with NT1 showed satisfying PA levels despite their daytime sleepiness. LTPA engagement was associated with higher quality of life. An APA intervention could be effective in children with narcolepsy, especially for those with depressive feelings.

Unveiled central hypoventilation after tracheotomy in anti-IgLON5 disease: a case report.

Anti-IgLON5 disease is a recently described entity that has been associated with neurological symptoms and sleep disturbances including sleep breathing disorders. Sleep stridor as well as obstructive and less often central sleep apnea have been reported but rarely needing ventilation on tracheotomy. We report the case of a patient in whom obstructive sleep apnea with secondary development of dysphagia and recurrent aspiration pneumonia led to the diagnosis of anti-IgLON 5 disease. Acute respiratory failure due to laryngospasm required intubation and eventually tracheotomy. Yet hypoventilation persisted, and polysomnography demonstrated central sleep apnea alternating with sleep-related tachypnea. Nocturnal ventilation was thus reintroduced. The association of obstructive sleep apnea with dysphagia is a potential red flag for anti-IgLON5 disease, which remains an overlooked diagnosis. Breathing disorders can be complex in this context, with a mixed obstructive and central pattern whose central component can be unveiled after tracheotomy. This highlights the importance of closely monitoring sleep and respiration even after tracheotomy. CITATION: Tankéré P, Le Cam P, Folliet L, et al. Unveiled central hypoventilation after tracheotomy in anti-IgLON5 disease: a case report. J Clin Sleep Med. 2023;19(9):1701-1704.

Sleep disturbances in early clinical stages of psychotic and bipolar disorders: A meta-analysis.

OBJECTIVE: To provide a qualitative view and quantitative measure of sleep disturbances across and between early stages - clinical ultra high-risk and first episode - of psychotic and bipolar disorders. METHODS: Electronic databases (PubMed, Cochrane, Embase, PsychINFO) were searched up to March 2021 for studies comparing sleep measures between individuals with an early stage and controls. Standard mean deviations (Cohen's d effect sizes) were calculated for all comparisons and pooled with random-effects models. Chi-square tests were used for direct between-subgroups (ultra high-risk vs first episode) comparisons of standard mean deviations. The effects of age, sex ratio, symptoms and treatment were examined in meta-regression analyses. RESULTS: A database search identified 13 studies that contrasted sleep measures between individuals with an early stage (N = 537) and controls (N = 360). We observed poorer subjective sleep quality (standard mean deviation = 1.32; 95% confidence interval, [1.01, 1.62]), shorter total sleep time (standard mean deviation =-0.44; 95% confidence interval, [-0.67, -0.21]), lower sleep efficiency (standard mean deviation = -0.72; 95% confidence interval, [-1.08, -0.36]), longer sleep onset latency (standard mean deviation = 0.75; 95% confidence interval, [0.45, 1.06]) and longer duration of wake after sleep onset (standard mean deviation = 0.49; 95% confidence interval, [0.21, 0.77]) were observed in early stages compared to controls. No significant differences were observed for any of the reported electroencephalographic parameters of sleep architecture. No significant between-subgroups differences were observed. Meta-regressions revealed a significant effect of the age and the antipsychotic status on subjective measures of sleep. CONCLUSION: The early stage population presents with significant impairments of subjective sleep quality continuity, duration and initiation. Systematic assessments of sleep in early intervention settings may allow early identification and treatment of sleep disturbances in this population.