Serum melatonin kinetics and long-term melatonin treatment for sleep disorders in Rett syndrome
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Fig. 1
Sleep-wake periods as observed and recorded by the mother of patient 1 before melatonin treatment (A) and during treatment with oral melatonin, 5 mg/day (B).
Fig. 2
Serum melatonin pattern before (closed circle) and during (open circle) melatonin treatment in patient 1. Oral melatonin, 5 mg administration at 1930 h. Sleep time before (closed box) and during (stippled box) melatonin treatment. Serum melatonin levels in normal prepubertal children reported by Ehrenkranz et al. [11] (shaded box).
Fig. 3
Sleep-wake periods as observed and recorded by the mother of patient 2 before melatonin treatment (A) and during treatment with oral melatonin, 5 mg/day (B).
Fig. 4
Serum melatonin pattern before (closed circle) and during (open circle) melatonin treatment in patient 2. Oral melatonin, 5 mg administration at 2000 h. Sleep time before (closed box) and during (stippled box) melatonin treatment. Serum melatonin levels in normal prepubertal children reported by Ehrenkranz et al. [11] (shaded box).
Abstract
We studied the circadian rhythm of serum melatonin levels in two patients with classical Rett syndrome having severe sleep disorders; serum melatonin levels were measured before and during melatonin treatment using radioimmnoassay. Patient 1 had a free-running rhythm of sleep-wake cycle from 3 years of age. At the age of 4 years, the peak time of melatonin was delayed 6 h compared to normal control and the peak value was at the lower limit. Patient 2 had a fragmented sleep pattern accompanied by night screaming from 1 year and 6 months of age. At the age of 10 years, the peak time of melatonin secretion was normal but the peak value was at the lower limit. These patients were given 5 mg melatonin orally prior to bedtime. Exogenous melatonin dramatically improved the sleep-wake cycle in patient 1. In patient 2, exogenous melatonin showed a hypnotic effect but early morning awakenings occurred occasionally. When melatonin treatment was stopped, the sleep disorders recurred and re-administration of 3 mg melatonin was effective in both patients. The effect was maintained over 2 years without any adverse effects. These findings suggests that sleep disorders in patients with Rett syndrome may relate with an impaired secretion of melatonin.
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