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Subjective sleep disturbances and circadian rhythm characteristics are associated with specific behavioral difficulties in children with Smith-Magenis syndrome, despite ongoing treatment.

In a cohort of 20 children with Smith-Magenis syndrome (mean age 10 ± 2.5 years), 70% had pathological sleep disturbances, and actigraphy showed reduced sleep efficiency in 88% and early L5 onset in 92%. Subjective sleep disturbances (insomnia, non-restorative sleep) correlated with irritability, social withdrawal, stereotyped behaviors, and inappropriate speech, while an earlier L5 onset (circadian) was associated with greater social withdrawal and hyperactivity. No significant associations were found between objective sleep measures and behaviors.

Last updated: Aug 31, 2026•0 RCTs•📖 Read as article →

Evidence Score

Evidence Score34/100
Human RCT☆☆☆☆☆
Meta-analysis☆☆☆☆☆
Mechanism★★★★★
Safety★★★★☆
Confidencelow

Study Evidence

Study 1. Potential Contributions of Sleep and Circadian Rhythms to Behavioral Difficulties in Children with Smith-Magenis Syndrome in Real Life: An Actigraphy-Based Study.

observational

Comajuan M, Plancoulaine S, Demily C, Babinet MN, Lioret J, Brunel L, Rivet L, Schröder CM, Guyon A, Franco P · Children (Basel, Switzerland) (2026)

Participants: N/A
Duration: 15-day actigraphy, cross-sectional
Intervention: Observational study; no intervention tested. Sleep and circadian rhythms were assessed via questionnaires (SDSC, H&O) and 15-day actigraphy; behaviors via Aberrant Behavior Checklist-Community (ABC-C).
Outcome: Behavioral difficulties (ABC-C subscales: irritability, social withdrawal, stereotyped behaviors, hyperactivity, inappropriate speech) and their correlations with sleep/circadian measures.
Effect Size: Correlation coefficients: insomnia-irritability r=0.472, insomnia-social withdrawal r=0.701, insomnia-stereotyped behaviors r=0.648, non-restorative sleep-inappropriate speech r=0.476, morningness-irritability r=-0.565, L5 onset-social withdrawal r=-0.556, L5 onset-hyperactivity r=-0.560
Population: Children aged 5-13 years with genetically confirmed Smith-Magenis syndrome (75% 17p11.2 microdeletion, 25% RAI1 mutation), 55% female, 95% on melatonin.

Result:

PubMed: 42650327DOI: 10.3390/children13080987

Mechanism Graph

Sleep disturbances (e.g., insomnia, non-restorative sleep) may exacerbate emotional and behavioral regulation difficulties in children with SMS.
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Circadian rhythm phase (early L5 onset) may influence activity levels and social interaction, contributing to hyperactivity and withdrawal.
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Subjective sleep complaints reflect perceived sleep quality, which may be more closely tied to behavioral outcomes than objective actigraphy measures.
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Result: Distinct associations between subjective sleep/circadian parameters and specific behavioral domains, suggesting potential targets for intervention.

Limitations

  • ⚠Small sample size (n=20) and exploratory secondary analysis, limiting generalizability.
  • ⚠Cross-sectional design prevents causal inference; associations may be bidirectional.
  • ⚠High use of concomitant medications (melatonin, psychostimulants, beta-blockers) may confound sleep and behavior measures.
  • ⚠Objective actigraphy measures did not correlate with behaviors, suggesting possible measurement issues or lack of sensitivity.

Frequently Asked Questions

What sleep disturbances are common in children with Smith-Magenis syndrome?▼

In this study, 70% had pathological sleep disturbances, including insomnia (50%) and excessive daytime sleepiness (40%). Actigraphy showed reduced total sleep time in 71%, reduced sleep efficiency in 88%, and long wake after sleep onset in 59%.

Are sleep problems associated with specific behavioral difficulties in SMS?▼

Yes, subjective sleep disturbances like insomnia were positively correlated with irritability, social withdrawal, and stereotyped behaviors. Non-restorative sleep was associated with inappropriate speech. Circadian phase (earlier L5 onset) was associated with more social withdrawal and hyperactivity.

Did objective sleep measures (actigraphy) correlate with behavior?▼

No significant associations were found between objective sleep measures (e.g., total sleep time, sleep efficiency) and any behavioral subscale, suggesting that subjective sleep complaints may be more relevant to behavior in this population.

What is the clinical implication of these findings?▼

The findings suggest that addressing subjective sleep complaints and circadian rhythm phase (e.g., morning chronotype) might help manage behavioral difficulties in children with SMS, but confirmatory studies are needed.

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References

  1. 1.Comajuan M, Plancoulaine S, Demily C, Babinet MN, Lioret J, Brunel L, Rivet L, Schröder CM, Guyon A, Franco P. "Potential Contributions of Sleep and Circadian Rhythms to Behavioral Difficulties in Children with Smith-Magenis Syndrome in Real Life: An Actigraphy-Based Study.." Children (Basel, Switzerland), 2026. PMID: 42650327 DOI: 10.3390/children13080987
Disclaimer: This content is for educational purposes only and is not medical advice. Evidence scores reflect the quality and quantity of available research, not clinical recommendations. Always consult a healthcare professional before starting any supplement or intervention.