Rest-Activity Disturbances in Children with Septo-Optic Dysplasia Characterized by Actigraphy and 24-Hour Plasma Melatonin Profiles

• Published in: The journal of clinical endocrinology and metabolism 2010-10, Vol.95 (10), p.E198-E203
• Main Authors: Webb, E. A, O'Reilly, M. A, Orgill, J, Dale, N, Salt, A, Gringras, P, Dattani, M. T
• Format: Article
• Language: English
• Subjects: Actigraphy - methods; Activity Cycles - physiology; Child; Child, Preschool; Chronobiology Disorders - blood; Chronobiology Disorders - complications; Chronobiology Disorders - diagnosis; Chronobiology Disorders - physiopathology; Circadian Rhythm; Diagnostic Techniques, Endocrine; Female; Humans; Infant; Male; Melatonin - analysis; Melatonin - blood; Melatonin - metabolism; Metabolome; Rest - physiology; Septo-Optic Dysplasia - blood; Septo-Optic Dysplasia - complications; Septo-Optic Dysplasia - diagnosis; Septo-Optic Dysplasia - physiopathology
• ISSN: 0021-972X; 1945-7197
• DOI: 10.1210/jc.2010-0027

Introduction: A trial of melatonin treatment in children with septo-optic dysplasia (SOD) and sleep disruption is accepted clinical practice in many centers. However, no objective measurements of sleep/activity patterns with 24-h melatonin profiles have been published for these individuals, and the pathophysiological basis underlying sleep disorders in SOD remains largely unknown. Methods: We studied six children with rest-activity disturbances and SOD. All wore an Actiwatch-Mini (a noninvasive method of detecting and recording movement intensity) for 2 wk and were admitted to hospital for a 24-h period during which hourly measurements of serum melatonin were taken. Sleep data were analyzed in conjunction with a detailed sleep diary. Ethical approval was obtained for these studies. Results: Two children produced virtually no melatonin throughout the 24-h period of measurement and had fragmented sleep patterns with no evidence of a non-24-h sleep-wake disorder or delayed sleep-phase disorder. One child had a normal melatonin profile despite actigraphy showing an arrhythmic sleep pattern. The remaining three children had fragmented sleep, with two having normal melatonin profiles and one having a modest increase in daytime melatonin concentrations, making the timing of dim-light melatonin onset difficult to discern. Conclusions: There is considerable variation in timing and amount of melatonin secretion in these children. Surprisingly, none of the children had either actigraphic or melatonin profile evidence of a non-24-h sleep-wake disorder or delayed sleep-phase disorder. Understanding the heterogeneous nature of underlying sleep disorders in this group of children is important and has implications for their management. Melatonin secretion in septo-optic dysplasia is frequently abnormal; however, abnormalities are not associated with evidence of a non-24-hour sleep-wake disorder, or delayed sleep-phase disorder.