Sabine Plancoulaine

Background: Smith–Magenis syndrome (SMS) is a rare neurodevelopmental disorder characterized by severe sleep disturbances, circadian rhythm dysregulation, and prominent behavioral difficulties. Objective: To examine associations between subjective and objective sleep and circadian characteristics and behavioral difficulties in children with SMS in real life. Methods: Children aged 5–13 years with genetically confirmed SMS were included in a prospective single-center cohort study. Sleep and circadian rhythms were assessed using parent-reported questionnaires (Sleep Disturbance Scale for Children (SDSC), Horne and Östberg Morningness–Eveningness Questionnaire (H&O)) and 15-day home-based wrist actigraphy. Behavioral difficulties were evaluated using the Aberrant Behavior Checklist-Community (ABC-C). Associations between subjective and objective sleep and circadian measures with behavioral difficulties were studied using Spearman correlations for each ABC-C subscale. Results: Among the 20 children included (55% female, mean age 10 ± 2.5 years), 75% were carriers of a 17p11.2 microdeletion and 25% had an RAI1 mutation. Overall, 95% of children were treated with melatonin, 55% with psychostimulants and 20% with beta-blockers. According to the SDSC results, pathological sleep disturbances were present in 14/20 (70%) children and concerned mainly complaints of insomnia (10/20, 50%) and excessive daytime sleepiness (8/20, 40%). The H&O chronotypes confirmed the morning type in 70% of patients. Sleep and circadian measures obtained by actigraphy showed reduced total sleep time in 71% of the children, reduced sleep efficiency in 88%, long wake after sleep onset (WASO > 60 min) in 59%, as well as an early L5 onset (5 h period with the least movements < 23:38) and M10 onset (10 h period with the highest activity levels < 09:12) in 92% and 62% of children, respectively. The behavioral domains of the ABC-C with the highest scores were hyperactivity (mean 52.1, range 2.1–97.9) and irritability, agitation, and crying (mean 46.9, range 17.8–82.2). In the analysis of the relationship between behavior and sleep, a positive correlation was found between sleep disorders and stereotyped behaviors (r = 0.472, p = 0.048), insomnia and irritability, social withdrawal and stereotyped behaviors (r = 0.472, p = 0.048; r = 0.701, p = 0.001; and r = 0.648, p = 0.004, respectively), and non-restorative sleep and inappropriate speech (r = 0.476, p = 0.046). No significant association was found between objective sleep measures and behaviors. While subjective assessments suggested that a stronger morning chronotype was associated with lower irritability (r = −0.565, p = 0.015), actigraphy-derived circadian rhythm analyses revealed that an earlier L5 onset was associated with greater social withdrawal and hyperactivity (r = −0.556, p = 0.048 and r = −0.560, p = 0.049, respectively). Conclusions: The present cohort study of children with SMS studied in real-life conditions, using both objective and subjective measures, shows that patients continued to experience sleep and behavioral disturbances despite treatment. Exploratory analyses identified distinct associations between subjective sleep disturbances, circadian rhythm characteristics, and specific behavioral domains. While subjective sleep disturbances were associated with irritability, stereotypic behavior, and inappropriate speech, circadian rhythm parameters are specifically associated with hyperactivity. Both were also associated with social withdrawal. As these findings arise from an exploratory observational secondary analysis, they should be considered hypothesis-generating and require confirmation in larger prospective studies. If confirmed, they may help identify sleep and circadian rhythm characteristics as potential targets for future interventions in children with SMS.

This study examines the transition from biphasic to monophasic sleep in preschool children aged 3–5 years by characterising nap behaviours. Parent‐reported data from 1498 preschool children in France were analysed using latent class analysis, considering nap frequency, duration, timing and acceptance at home and school. Five distinct profiles were identified: regular, frequent, school, home and non‐nappers. While several profiles followed an expected age‐related pattern, two reflected discrepancies between home and school nap practices, suggesting the influence of environmental constraints beyond biological maturation. Context‐dependent profiles showed signs of sleep misalignment: school nappers exhibited more sleep onset difficulties, whereas older home nappers showed greater daytime fatigue despite normal nighttime sleep, suggesting unmet sleep needs. Together, these findings indicate that nap cessation reflects a dynamic interplay between developmental maturation and contextual factors, rather than age alone. The results support flexible and individualised preschool nap practices, rather than uniform policies based solely on chronological age.

Background/Objectives: Comorbid insomnia and sleep apnea (COMISA) in children is poorly documented. This study aimed to evaluate the frequency of COMISA and to explore its clinical and polysomnographic characteristics in children referred for polysomnography (PSG) for any sleep complaint. Methods: All patients with a complete insomnia sub-score on the Sleep Disturbance Scale for Children (SDSC; for children from 6 months to 16 years old) who underwent a night PSG in a pediatric sleep unit (2018–2024) were included in this retrospective study. Pathological SDSC insomnia sub-score defined insomnia and obstructive apnea-hypopnea index ≥ 2/h on PSG defined OSA. Questionnaires regarding sleepiness, depression, anxiety, and hyperactivity were also collected. Results: Children had isolated insomnia in 11.5% of cases, isolated OSA in 37.5%, and COMISA in 13.5%. Insomnia frequency was not different between patients with and those without OSA (26.5% vs. 23.5%). COMISA was more frequent in patients under 4 years old than in older ones (39.1% vs. 5.8%). No polysomnographic or clinical characteristic of COMISA was identified, except that OAHI was higher in children with isolated OSA. Patients with COMISA or isolated insomnia were more anxious than those with isolated OSA. Conclusions: Unlike in adults, the present findings do not support a mutual association between OSA and insomnia in children. OSA severity was lower in children with COMISA. Anxiety levels were higher in children with insomnia, regardless of the presence of OSA, suggesting that anxiety should be assessed in all children with OSA.

Sleep troubles and respiratory and allergic health issues are associated in children, but the timeline of their association is overlooked. This study investigates the associations between sleep patterns at age 1 and respiratory and allergic multi‐trajectories (RespA‐MTG) between ages 1 and 5.5, and the associations between these multi‐trajectories and sleep at age 5.5 in the ELFE birth cohort. Sleep clusters at ages 1 and 5.5 (based on nocturnal and diurnal sleep duration, sleep onset difficulties, and night awakenings) and RespA‐MTG between ages 1 and 5.5 (based on wheezing, asthma medication, eczema, allergic conjunctivitis) were identified using data‐driven methods. Associations between sleep clusters and RespA‐MTG were assessed using multinomial regressions adjusted for confounders in 9577 children. Two sleep clusters were identified at ages 1 and 5.5: good sleepers (79.9% at age 1, 83.1% at 5.5) and poor sleepers (20.1% and 16.9%, respectively). Four RespA‐MTG were identified: pauci‐symptomatic (44.4%), persistent non‐respiratory allergic symptoms (23.1%), transient early respiratory symptoms (25.2%), and persistent respiratory and allergic symptoms (7.3%). Poor sleep at age 1 was associated with higher odds of transient early respiratory symptoms (Odds Ratio [95% Confidence Interval], 1.14 [0.99–1.31]) and persistent respiratory and allergic symptoms (1.29 [1.05–1.59]). Results were reinforced in children without wheezing at 2 months. A borderline association was observed between persistent respiratory and allergic symptoms and sleep at 5.5 in good sleepers at 1 year (1.22 [0.98–1.50]). In conclusion, sleep disturbances at age 1 are associated with later poorer respiratory and allergic health, suggesting early sleep troubles may predict these health concerns.

Background/Objectives: Sleep is essential for children’s well-being, yet insufficient sleep duration and quality are common among preschoolers. The brain–gut microbiota axis, a bidirectional communication network connecting the brain, the gastrointestinal tract, and the microorganisms living there, known as the gut microbiota, influences sleep regulation, but its role in children remains largely unexplored. Here, we examined the association between gut microbiota and sleep in preschoolers from Étude Longitudinale Française depuis l’Enfance (ELFE) birth cohort study. Methods: This study included 597 children (51.2% boys) with available stool samples and sleep data at 3.5 years. The gut microbiota data was analyzed using bacterial 16S rRNA sequencing. Data on day and night sleep durations and frequencies of sleep onset difficulties and night waking were collected through telephone questionnaires and grouped into ‘optimal’ and ‘suboptimal’ clusters using Latent Class Analysis. Statistical analyses involved multivariate logistic regressions or multivariate permutation analysis of variance, controlling for confounders. Results: In total, 25% of the included children were in the suboptimal sleep cluster. No significant associations were found between gut microbiota diversity and composition and sleep clusters at age 3.5 years. Similarly, no differences were found in the abundance of specific microbiota genera between the two sleep clusters. Conclusions: While emerging evidence suggests correlations between gut microbiota and sleep in preschool children, our results do not confirm such correlations. The data used in this study were obtained from a homogeneous, high socioeconomic population, which must be considered when interpreting the findings. Further research is needed to validate the results of this study.

To examine the association between intimate partner psychological violence (P-IPV) from before pregnancy to 2 years after the child’s birth and child’s sleep patterns, i.e. sleep onset difficulty (SOD), nighttime awakenings (NA), and nighttime sleep duration (NSD) between 2 and 5 years of child’s age. Data come from the population-based French birth ELFE cohort launched in 2011. P-IPV was assessed before and during pregnancy, at 2 months and 2 years post-partum. Children’s sleep patterns were measured at 2, 3, and 5 years of age. Group-based trajectory modelling was used to identify trajectories of P-IPV and each child’s sleep patterns. Associations between P-IPV and children’s sleep trajectories were assessed by weighted multivariate logistic regressions. Five P-IPV trajectories were identified: minimal (64%), prenatal (14%), decreasing (9%), increasing (8%), and persistent (5%). Two trajectories of SOD (few 65% and many 35%), three trajectories of NA (few 49%, decreasing 24%, and many 23%), and three trajectories of NSD (short 21%, medium 56%, and long 23%) were identified. About 9513, 9512, and 9499 children were included in comparative analyses, respectively, focused on SOD, NA, and NSD. Increasing and persistent P-IPV trajectories were both associated with the trajectory of many SODs [odds ratio (OR) = 1.53, 95% confident interval (CI) = 1.24–1.91; and OR = 1.71, 95% CI = 1.31–2.22, respectively] and the trajectory of many NA (OR = 1.66, 95% CI = 1.29–2.13); and (OR = 1.95, 95% CI = 1.42–2.69, respectively). Associations between persistent P-IPV and decreasing and many NA were significant among girls (OR = 1.76, 95% CI = 1.12–2.75 and OR = 2.27, 95% CI = 1.39–3.71, respectively), but not among boys. Family interventions in response to IPV should pay particular attention to sleep patterns of children exposed to IPV.

To assess the association between sleep irregularity, anxiety, and depression while controlling for other sleep dimensions and using a longitudinal design. Longitudinal cohort study which started in April 2020 during the first French lockdown in the general population. Follow-up questionnaires were completed in June 2020, a period without lockdown measures. Participants were asked about their sleep (regularity, duration, timing, complaints) and their anxiety (General Anxiety Disorder-7) and depressive (Patient Health Questionnaire-9) symptoms. A total of 3745 participants were included (mean age: 28.9 years) with 2945 women (78.6%). At baseline, 38.1% (1428) of participants reported irregular sleep timing, 23.8% (891) anxiety and 28.9% (1081) depressive symptoms. In cross-sectional analyses, irregular sleep timing was associated with a 2.5-fold higher likelihood of anxiety and a 4-fold higher likelihood of depressive symptoms compared to regular sleepers. Associations were not explained by the other sleep dimensions and persisted in a longitudinal analysis, with irregular sleep timing at baseline being associated with anxiety (OR = 3.27[1.58-6.76]) and depressive symptoms (OR = 3.45[1.66-7.19]) during follow-up. The results show a strong association between sleep irregularity and mental health. Furthers studies are needed to explore how sleep regularity could promote good mental health in non-clinical populations.