Melatonin excretion in normal children and in tuberous sclerosis complex with sleep disorder responsive to melatonin
Circadian Rhythm; Male; Case-Control Studies; Child; Humans; Adolescent; Female; Child Preschool; Administration Oral; Reference Values; Tuberous Sclerosis; Antioxidants; Melatonin; Sleep Wake Disorders; Antioxidants/pk [Pharmacokinetics]; Circadian Rhythm; Melatonin/aa [Analogs & Derivatives]; Melatonin/pk [Pharmacokinetics]; Sleep Wake Disorders/co [Complications]; Tuberous Sclerosis/co [Complications]; 0 (Antioxidants); 2208-40-4 (6-sulfatoxymelatonin); Antioxidants/ad [Administration & Dosage]; JL5DK93RCL (Melatonin); Melatonin/ad [Administration & Dosage]; Melatonin/ur [Urine]; Sleep Wake Disorders/pp [Physiopathology]; Tuberous Sclerosis/pp [Physiopathology]; sleep disturbance/disorders; trajectory; characteristics; melatonin
To determine normal melatonin excretion patterns in healthy children without sleep disorder and to compare these with those of patients with tuberous sclerosis complex and sleep disorder responsive to exogenous melatonin, we measured 6-sulfatoxymelatonin excretion in 21 healthy children and in 7 patients with tuberous sclerosis complex and sleep disorder responsive to melatonin (a 5 mg oral dose increasing total sleep time). Total excretion, cosinor percentage, and acrophase time of 6-sulfatoxymelatonin excretion were estimated. In normal children, total 6-sulfatoxymelatonin excretion was range 11.1 to 40.2 microg (mean 19.0 microg, SD 7.4 microg); cosinor percentage rhythm range was 52.9% to 100% (mean 87%, median 94%); and acrophase time range was 23 hours, 54 minutes to 10 hours, 42 minutes (mean 5 hours, 54 minutes; median 4 hours, 12 minutes). Fifth and 95th percentiles were 11.1 to 29.0 microg, 57.8% to 99.9%, and 2 hours, 1 minute to 10 hours, 4 minutes. In tuberous sclerosis, normal patterns of melatonin excretion were seen in responders. Circadian patterns of melatonin excretion were similar in children and adults. We propose that exogenous melatonin can act by a simple sedative action.
Hancock E; O'Callaghan F; English J; Osborne J P
Journal of Child Neurology
2005
Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
<a href="http://doi.org/10.1177/08830738050200010301" target="_blank" rel="noreferrer noopener">10.1177/08830738050200010301</a>
Melatonin ineffective in neuronal ceroid lipofuscinosis patients with fragmented or normal motor activity rhythms recorded by wrist actigraphy
Adolescent; Adult; Antioxidants/tu [Therapeutic Use]; Child; Circadian Rhythm; Dose-Response Relationship; Drug Electrophysiology; Female; Humans; Male; Melatonin/tu [Therapeutic Use]; Neuronal Ceroid-Lipofuscinoses/dt [Drug Therapy]; Sleep Wake Disorders/th [Therapy]; 0 (Antioxidants); JL5DK93RCL (Melatonin); sleep disturbance/disorders; NCL3; pharmacologic intervention; melatonin
Melatonin was tested as a sleeping pill in five patients with neuronal ceroid lipofuscinoses. The single-blind, placebo-controlled study consisted of motor activity recordings, sleep logs, and administration of placebo or melatonin (2.5 or 5 mg). Daily motor activity rhythms were measured by wrist actigraphy during four 7-day periods (baseline, placebo, melatonin 2.5 mg, and melatonin 5 mg). The placebo or melatonin was administered in the evenings for 3 weeks, and the recordings were made during the last week of the 3-week treatment. Sleep logs were kept by the caregivers during the recordings. Based on period analyses, the activity recordings were evaluated to display a normal (24-h) or fragmented rhythm. Three patients had normal motor activity patterns during the baseline recordings, and administration of placebo or melatonin did not affect their rest/activity rhythms. Two patients had abnormally fragmented activity rhythms during the baseline periods, and administration of placebo or melatonin did not induce synchronization. According to the actigraphic data, there were no changes in activity rhythms resulting from administration of melatonin. However, based on the observations, three families reported that melatonin slightly improved the sleep quality of the patients. These controversial findings show the difficulties involved in specifying the role of melatonin in modulating sleep. Thus, we conclude that more evidence is required before the significance of melatonin as a sleeping pill is defined.Copyright 1999 Academic Press.
Hätönen T; Kirveskari E; Heiskala H; Sainio K; Laakso M L; Santavuori P
Molecular Genetics and Metabolism
1999
Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
<a href="http://doi.org/10.1006/mgme.1999.2815" target="_blank" rel="noreferrer noopener">10.1006/mgme.1999.2815</a>
Assessment of sleep in children with mucopolysaccharidosis type III
Actigraphy; Adolescent; Case-Control Studies; Child; Preschool; Female; Humans; Male; Melatonin/me [Metabolism]; Mucopolysaccharidosis III/pp [Physiopathology]; Sleep/ph [Physiology]; Time Factors; JL5DK93RCL (Melatonin); sleep disturbance/disorders; MPSIIIA; MPSIIIB; trajectory; characteristics
Sleep disturbances are prevalent in mucopolysaccharidosis Type III (MPS III), yet there is a lack of objective, ecologically valid evidence detailing sleep quantity, quality or circadian system. Eight children with MPS III and eight age-matched typically developing children wore an actigraph for 7-10 days/nights. Saliva samples were collected at three time-points on two separate days, to permit analysis of endogenous melatonin levels. Parents completed a sleep questionnaire and a daily sleep diary. Actigraphic data revealed that children with MPS III had significantly longer sleep onset latencies and greater daytime sleep compared to controls, but night-time sleep duration did not differ between groups. In the MPS III group, sleep efficiency declined, and sleep onset latency increased, with age. Questionnaire responses showed that MPS III patients had significantly more sleep difficulties in all domains compared to controls. Melatonin concentrations showed an alteration in the circadian system in MPS III, which suggests that treatment for sleep problems should attempt to synchronise the sleep-wake cycle to a more regular pattern. Actigraphy was tolerated by children and this monitoring device can be recommended as a measure of treatment success in research and clinical practice.
Mahon L V; Lomax M; Grant S; Cross E; Hare D J; Wraith J E; Jones S; Bigger B; Langford-Smith K; Canal M
PLoS ONE
2014
Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
<a href="http://doi.org/10.1371/journal.pone.0084128" target="_blank" rel="noreferrer noopener">10.1371/journal.pone.0084128</a>