title: A combined behavioral/pharmacological treatment of sleep-wake schedule disorder in Angelman syndrome.
abstract: Angelman syndrome (AS) is a genetic disorder associated with a deletion on chromosome 15. Behavior problems among children with AS include sleep difficulties. Data are presented on the successful treatment of a sleep-wake schedule disorder (SWSD) in a 9-year-old boy with AS. The treatment program included behavioral and pharmacological components. During baseline, the child slept a mean of 1.9 hours per night and 1.3 hours during the day; night sleep was increased to a mean of 8.3 hours and day sleep was reduced to a mean of .08 hours after introduction of the full-treatment program. Medication was discontinued subsequently, and the child slept a mean of 7.8 hours during the night and .07 hours during the day. At 45-day follow-up, night sleep was maintained at 7.1 hours and day sleep remained stable at .29 hours. This is the first known report of an effective treatment of a SWSD in a child with AS.
authors.affiliation: Department of Behavioral Psychology, Kennedy Krieger Institute, Baltimore, MD 21205.
authors.forename: J A
authors.initials: JA
authors.lastname: Summers
authors.forename: P S
authors.initials: PS
authors.lastname: Lynch
authors.forename: J C
authors.initials: JC
authors.lastname: Harris
authors.forename: J C
authors.initials: JC
authors.lastname: Burke
authors.forename: D B
authors.initials: DB
authors.lastname: Allison
authors.forename: L
authors.initials: L
authors.lastname: Sandler
mesh_terms: D001521:Behavior Therapy; D002648:Child; D002872:Chromosome Deletion; D002884:Chromosomes, Human, Pair 15; D002940:Circadian Rhythm / Q000187:drug effects / Q000235:genetics*; D003131:Combined Modality Therapy; D004155:Diphenhydramine / Q000627:therapeutic use*; D006801:Humans; D008607:Intellectual Disability / Q000235:genetics* / Q000523:psychology / Q000628:therapy; D008297:Male; D008831:Microcephaly / Q000235:genetics* / Q000523:psychology; D011930:Reaction Time / Q000187:drug effects; D012893:Sleep Wake Disorders / Q000235:genetics* / Q000523:psychology / Q000628:therapy
chemical_list: D004155:Diphenhydramine