beta(1)-adrenergic antagonists improve sleep and behavioural disturbances in a circadian disorder, Smith-Magenis syndrome |
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Authors: | De Leersnyder H de Blois M C Vekemans M Sidi D Villain E Kindermans C Munnich A |
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Affiliation: | Department of Genetics and Unit INSERM 393, Hôpital Necker Enfants-Malades, 149 rue de Sevres, 75743 Paris Cedex 15, France. |
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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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Keywords: | Smith-Magenis syndrome circadian rhythms melatonin |
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