Objective To compare symptoms and sleep characteristics in patients diagnosed with narcolepsy‐cataplexy (NC) before and after the age of 18 years. Methods De novo patients with NC diagnosis completed a standardized questionnaire and interview, followed by a sleep study. The clinical and sleep measures were compared between patients diagnosed before (46 children, median age: 12 year old) and after (46 adults, median age: 28.5 year old) 18 years of age. Results The frequency of obesity (54% vs 17%), night eating (29% vs 7%), parasomnia (89% vs 43%), sleep talking (80% vs 34%), and sleep drunkenness (69% vs 24%) were higher in children than in adults, the frequency of sleep paralysis was lower (20% vs 55%) but the frequency of cataplexy and the severity of sleepiness were not different. Children scored higher than adults at the attention‐deficit/hyperactivity disorder (ADHD) scale. Depressive feelings affected not differently children (24%) and adults (32%). However, adults had lower quality of life than children. There was no difference between groups for insomnia and fatigue scores. Quality of life was essentially impacted by depressive feelings in both children and adults. Obstructive apnea‐hypopnea index (OAHI) was lower in children with higher mean and minimal oxygen saturation than in adults. No between‐group differences were found at the multiple sleep latency test. The body mass index (z‐score) was correlated with OAHI (r = .32). Conclusion At time of NC diagnosis, children have more frequent obesity, night eating, parasomnia, sleep talking, drunkenness, and ADHD symptoms than adults, even if sleepiness and cataplexy do not differ. These differences should be considered to ensure a prompt diagnosis.
The involvement of sleep in cognitive functioning is well known, but only a few studies have examined objective sleep parameters in children with high intellectual potential (HP). The main objective of this study was to compare sleep characteristics of 33 children with high intellectual potentialities (HP) (median 10 years old, 64% of boys) compared to 25 controls (median 11 years old, 64% of boys) and assess the difference between children with a homogeneous vs. a heterogeneous intelligence quotient (IQ) (i.e., a difference ≥15 points between verbal and non-verbal IQ). All children underwent a one-night polysomnography, an evaluation of intellectual quotient (IQ) and filled standardized questionnaires. Using non-parametric tests to compare groups’ characteristics, we found that children with HP had more heterogeneous IQ, more rapid eyes movement (REM) sleep and tended to have less stage 1 sleep than controls. They also had more insomnia and sleep complaints. The high amount of REM sleep in children with HP could be advantageous for learning and could partially explain their gift. This study highlights the necessity of investigating sleep disorders in children with HP during clinical routine and reinforces the hypothesis of the involvement of nocturnal sleep, and especially REM sleep, in daytime cognition and behavior.
High cognitive functioning could be a protective factor for school difficulties, behavioral and mood impairments in children with narcolepsy. To investigate this factor, we studied the intellectual abilities of 74 children with narcolepsy (43 boys, 11.7 years old at diagnosis, 91% of cataplexies, 64% obese, 100% HLA positive for DR-DQB1*06:02). All children underwent a one-night polysomnography followed by Multiple Sleep Latency Tests, an evaluation of intelligence quotient (IQ), and filled standardized questionnaires. Thirty-eight percent had high potentialities (HP defined by IQ > 130) and 48% had school difficulties. Using non-parametric tests, we found that HP children reported less difficulties at school and tended to have less impulsivity, conduct, and learning disorders than those without HP. They also tended to be less obese and had less desaturation. Using a multivariate regression analysis, we found an association between the REM sleep percentage and the IQ. REM sleep could be involved in the dynamic changes contributing to the equilibrium of intellectual functioning. This study highlights that despite their frequent school difficulties, narcolepsy per se is unlikely to be a cause of intellectual disability in children. Prompt diagnosis and management of comorbidities such as obesity and obstructive sleep apnea (OSA) could improve cognitive and school performances in these children.
Objectives To characterize the rapid weight gain (RWG) phenotype associated with the onset of childhood narcolepsy and to determine whether it could constitute a marker of severity of the disease. Methods RWG was defined using the BMI z‐score slope reported to one year (>0.67 SD) from symptom onset to disease diagnosis. We compared the clinical, metabolic, and sleep characteristics between patients with or without RWG at diagnosis. Pharmacological management, anthropometric, and clinical progression were also evaluated during the follow‐up. Results A total of 84 de novo narcoleptic pediatric patients were included; their median age at diagnosis was 12.0 years; 59.5% boys, 90.5% cataplexy, and 98.7% HLA‐DQB1*06:02, 57% had RWG profile. RWG patients were younger at diagnosis than non‐RWG patients, despite a shorter diagnostic delay. They had a higher BMI z‐score and a higher prevalence of obesity at diagnosis, but not at symptom onset, and higher adapted Epworth Sleepiness Scale and Insomnia Severity Index scores than non‐RWG patients. No differences on nocturnal polysomnography and multiple sleep latency tests were found between groups at disease diagnosis. After a median follow‐up of 5 years, RWG patients still had a higher BMI z‐score and a higher prevalence of obesity despite benefiting from the same therapeutic management and displaying improvement in sleepiness and school difficulties. Conclusions Narcoleptic RWG patients were younger, sleepier, and the prevalence of obesity was higher at diagnosis despite a shorter diagnostic delay than that of non‐RWG patients. These patients had also a higher risk of developing a long‐term obesity, despite a positive progression of their narcoleptic symptoms. RGW could then represent a maker of a more severe phenotype of childhood narcolepsy, which should inspire a prompt and more offensive management to prevent obesity and its complications.
Study objectives To determine the prevalence of metabolic syndrome (MS) in children with narcolepsy and to evaluate their clinical and sleep characteristics according to the different components of MS. Methods This retrospective study consisted of 58 de novo children with narcolepsy (median age: 12.7 y, 48.3% of boys). The recently published MS criteria in a French population of children were used. Clinical and sleep characteristics were compared between groups with different components of MS. Results MS was present in 17.2% of children with narcolepsy, among whom 79.3% presented with high homeostasis model assessment for insulin resistance (HOMA-IR), 25.9% with high body mass index (BMI), 24.1% with low high-density lipoprotein cholesterol (HDL-C), and 12.1% with high triglycerides. Patients with at least 2 MS components had more night eating behaviors and tended to have lower percentage of slow wave sleep (SWS) and more fragmented sleep. On multiple sleep latency test (MSLT), they had shorter mean sleep latencies to rapid eye movement (REM), non-rapid eye movement (NREM) sleep and tended to have more sleep onset REM periods (SOREMPs) than those with less than 2 MS components. Conclusions Insulin resistance was found to be the core metabolic disturbance in obese as well as in non-obese children with narcolepsy. Children with narcolepsy with at least 2 MS components presented a more severe daytime sleepiness and a higher prevalence of night eating behaviors than those with less than 2 MS components. Such children might benefit from early evaluation and management in order to prevent future complications.
ObjectiveA defect of the waking systems could constitute a factor of vulnerability for sudden infant death syndrome (SIDS). A decrease in orexin levels, which promotes wakefulness and activates histaminergic neurons (another hypothalamic wake-promoting system) has already been demonstrated between 2 and 6 months. This work aims to study the levels of histamine (HA), tele-methylhistamine (t-MeHA), its direct metabolite, and t-MeHA/HA ratio in the cerebrospinal fluid (CSF) of healthy children, to evaluate the maturation of the histaminergic system and its possible involvement in SIDS.MethodsSeventy Eight French children between 0 and 20 years (48.7% boys) were included, all of whom had a clinical indication for lumbar puncture, but subsequently found to be normal. Measurements of HA and t-MeHA in CSF were performed by reverse phase liquid chromatography coupled to mass spectrometry detection. Statistical analyses were performed using Spearman correlations and Non-parametric pairwise ranking tests.ResultsA negative correlation was found between age and CSF HA (r = −0.44, p < 10−4) and t-MeHA (r = −0.70, p < 10−4) levels. In pairwise comparisons, no difference in CSF HA and t-MeHA levels was observed between youngest age groups (i.e., 0–2 mo vs. 3–6 mo), but CSF HA and t-MeHA levels were significantly lower in older children (i.e., >6 mo vs. 0–6 mo). The CSF HA decrease with age was only observed in boys, who also presented global lower CSF HA levels than girls.ConclusionCSF HA and t-MeHA levels decrease with age in boys, and global levels are lower in boys than in girls. These results reveal changes in histaminergic transmission and metabolism during maturation. Whether lower CSF histamine values in boys compared to girls could contribute to their higher risk of SIDS warrants further research.
AimsTo compare the children's sleep electroencephalogram according to their intellectual profile.MethodsChildren were grouped according to their Wechsler Intelligence Scale for Children (WISC) scores (17 with normal intelligence quotient [IQ, NIQ] and 24 with high IQ [HIQ]). Comparisons of spectral power between groups and its relationship with WISC scores were assessed using analyses of variance and linear regression models, adjusted for age and sex.ResultsChildren with HIQ had more rapid eye movement (REM) sleep, especially late at night, and more power in slow‐frequency bands during REM sleep than those with NIQ. There were also positive associations between the processing speed index and the spectral power in β bands in NREM sleep, and with the spectral power in α, σ, β, and γ bands in REM sleep, with different associations between groups.ConclusionThe enhanced power in slow bands during REM sleep in children with HIQ overlaps with that of typical REM sleep oscillations thought to be involved in emotional memory consolidation. The dissimilar relationships between spectral power and WISC scores in NIQ and HIQ groups may underlie functional differences in brain activity related to cognitive efficiency, questioning the direction of the relationship between sleep and cognitive functioning.
Study Objectives Narcolepsy with cataplexy is associated with obesity in children. We proposed to assess whether metabolic complications were linked to narcolepsy regardless of obesity. The second aim of the study was to compare endocrine comorbidities in obese children with narcolepsy and control patients. Methods We performed a case-control study in Pediatric Sleep Unit and Pediatric Endocrinology Unit of Woman Mother Child Hospital (Lyon, France) comparing twenty-two children with narcolepsy with 22 sex-, pubertal stage-, and BMI-matched non-syndromic obese patients. Clinical examination, biological measurements including an oral glucose tolerance test (OGTT) and abdominal ultrasound were performed. Results No difference regarding glucidic, lipid profile, hepatic, respiratory and cardiovascular parameters were found between narcoleptic and control subjects. Insulin sensitivity did not differ between the two groups. Control patients had more first-degree family history of overweight or obesity than children with narcolepsy (83% vs 50%, p=0.05). Prevalence of precocious puberty in children with narcolepsy was not higher than in control subjects, but all the cases of advanced puberty involved children with narcolepsy who were diagnosed before 11 years old. All cases of central hypothyroidism belong to the narcoleptic group who presented lower TSH and fTA values compared to control children (respectively p=0.03 and p= 0.001). Conclusions No difference regarding metabolic complications was found between children with narcolepsy and control subjects. Thus, metabolic disorders may be related to weight gain rather than a narcolepsy specific risk. The presence of hypothyroidism and advanced puberty suggest a global involvement of hypothalamic structures in children with narcolepsy.
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