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Journal of Medical Genetics logoLink to Journal of Medical Genetics
. 2001 Sep;38(9):586–590. doi: 10.1136/jmg.38.9.586

β1-adrenergic antagonists improve sleep and behavioural disturbances in a circadian disorder, Smith-Magenis syndrome

H De Leersnyder 1, M-C de Blois 1, M Vekemans 1, D Sidi 1, E Villain 1, C Kindermans 1, A Munnich 1
PMCID: PMC1734944  PMID: 11546826

Abstract

Smith-Magenis syndrome (SMS) is a clinically recognisable contiguous gene syndrome ascribed to interstitial deletions of chromosome 17p11.2. Patients have a phase shift of their circadian rhythm of melatonin with a paradoxical diurnal secretion of the hormone. Serum melatonin levels and day-night behaviour were studied in nine SMS children (aged 4 to 17 years) given acebutolol, a selective β1-adrenergic antagonist (10 mg/kg early in the morning). Cardiac examination, serum melatonin, motor activity recordings, and sleep diaries were monitored before and after drug administration. The present study shows that a single morning dose of acebutolol suppressed the inappropriate secretion of melatonin in SMS. A significant improvement of inappropriate behaviour with increased concentration, delayed sleep onset, increased hours of sleep, and delayed waking were also noted. These results suggest that β1-adrenergic antagonists help to manage hyperactivity, enhance cognitive performance, and reduce sleep disorders in SMS.


Keywords: Smith-Magenis syndrome; circadian rhythms; melatonin

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Selected References

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