title: Effects of donepezil and serotonin reuptake inhibitor on acute regression during adolescence in Down syndrome.
abstract: A 14-year-old boy with Down syndrome (DS) showed a gradual decline in his daily activities and feeding capacities, and a marked deterioration triggered by a streptococcal infection was observed at the age of 15 years. He became bedridden, accompanied by sleep disturbance, sustained upward gaze, and generalized rigidity. Magnetic resonance imaging showed unremarkable findings, but antiglutamate receptor autoantibodies were positive in his cerebrospinal fluid. Treatment with thiamine infusion and steroid pulse therapy showed little effect, but gross motor dysfunction and appetite loss were ameliorated by the administration of l-DOPA and serotonin reuptake inhibitors. Thereafter, autistic behaviors predominated, including loss of social interaction, oral tendency, water phobia, and aggressiveness. Initiation of donepezil, an acetylcholinesterase inhibitor, resulted in the disappearance of these symptoms and total recovery of the patient to his previous psychosocial levels. We hypothesize that the acute regression in adolescence represents a process closely related to the defects of serotonergic and cholinergic systems that are innate to DS brains and not just a nonspecific comorbidity of depression or limbic encephalitis.
authors.affiliation: Division of Child Neurology, Institute of Neurological Science, Faculty of Medicine, Tottori University, Yonago, Japan. Electronic address: holbergs@nifty.com.
authors.forename: Akiko
authors.initials: A
authors.lastname: Tamasaki
authors.affiliation: Division of Child Neurology, Institute of Neurological Science, Faculty of Medicine, Tottori University, Yonago, Japan.
authors.forename: Yoshiaki
authors.initials: Y
authors.lastname: Saito
authors.affiliation: Division of Child Neurology, Institute of Neurological Science, Faculty of Medicine, Tottori University, Yonago, Japan.
authors.forename: Riyo
authors.initials: R
authors.lastname: Ueda
authors.affiliation: Division of Child Neurology, Institute of Neurological Science, Faculty of Medicine, Tottori University, Yonago, Japan.
authors.forename: Koyo
authors.initials: K
authors.lastname: Ohno
authors.affiliation: Division of Neuropsychiatry, Institute of Neurological Science, Faculty of Medicine, Tottori University, Yonago, Japan.
authors.forename: Katsutoshi
authors.initials: K
authors.lastname: Yokoyama
authors.affiliation: Division of Neuropsychiatry, Institute of Neurological Science, Faculty of Medicine, Tottori University, Yonago, Japan.
authors.forename: Takahiro
authors.initials: T
authors.lastname: Satake
authors.affiliation: Department of Brain Development and Neural Regeneration, Tokyo Metropolitan Institute of Medical Science, Tokyo, Japan.
authors.forename: Hiroshi
authors.initials: H
authors.lastname: Sakuma
authors.affiliation: National Epilepsy Center, Shizuoka Institute of Epilepsy and Neurological Disorder, Shizuoka, Japan.
authors.forename: Yukitoshi
authors.initials: Y
authors.lastname: Takahashi
authors.affiliation: Division of Developmental Disabilities, The Misakaenosono Mutsumi Developmental, Medical, and Welfare Center, Isahaya, Japan.
authors.forename: Tatsuro
authors.initials: T
authors.lastname: Kondoh
authors.affiliation: Division of Child Neurology, Institute of Neurological Science, Faculty of Medicine, Tottori University, Yonago, Japan.
authors.forename: Yoshihiro
authors.initials: Y
authors.lastname: Maegaki
mesh_terms: D000293:Adolescent; D001921:Brain / Q000503:physiopathology; D018450:Disease Progression; D000077265:Donepezil; D004314:Down Syndrome / Q000188:drug therapy* / Q000503:physiopathology; D004569:Electroencephalography; D006801:Humans; D007189:Indans / Q000627:therapeutic use*; D008297:Male; D018697:Nootropic Agents / Q000627:therapeutic use*; D010880:Piperidines / Q000627:therapeutic use*; D017367:Selective Serotonin Reuptake Inhibitors / Q000627:therapeutic use*; D016896:Treatment Outcome
chemical_list: D000077265:Donepezil; D007189:Indans; D018697:Nootropic Agents; D010880:Piperidines; D017367:Selective Serotonin Reuptake Inhibitors
keywords: Acetylcholine esterase inhibitor; Antiglutamate receptor antibody; Childhood; Deterioration; Down syndrome