The effects of low dose melatonin therapy on sleep behavior and serum melat
onin levels were studied in Angelman syndrome (AS) children suffering from
insomnia. 24-hour motor activity was monitored in 13 AS children (age 2-10
yr) in their home environments for 7 days prior to melatonin treatment and
for 5 days during which a 0.3 mg dose of melatonin was administered daily 0
.5-1 hour before the patient's habitual bedtime. Blood samples were withdra
wn at hourly intervals over two 21-hour periods in order to measure individ
ual endogenous serum melatonin levels and the levels induced by melatonin t
reatment, Actigraphic recording of motor activity, confirmed by parents' re
ports, showed a significant improvement in the patients' nocturnal sleep pa
ttern as a result of melatonin treatment. Analysis of the group data reveal
ed a significant decrease in motor activity during the total sleep period f
ollowing melatonin treatment, and an increase in the duration of the total
sleep period. Endogenous peak nocturnal melatonin values ranged from 19 to
177 pg/ml, The administration of melatonin elevated peak serum hormone leve
ls to 128-2800 pg/ml in children of different ages and body mass. These dat
a suggest that a moderate increase in circulating melatonin levels signific
antly, reduces motor activity during the sleep period in Angelman syndrome
children, and promotes sleep.