title: Use of subcutaneous flumazenil preparations for the treatment of idiopathic hypersomnia: A case report.
abstract: Idiopathic hypersomnia (IH) is a rare sleep disorder, recently hypothesized to be related to the production of a molecule that facilitates the binding of gamma-aminobutyric acid (GABA) to the GABA receptor. This paper reports on the treatment of a patient with IH who was treated with a 96-hour continuous low dose (4 mg/day) infusion of a benzodiazepine antagonist, flumazenil, followed by a slow-release flumazenil implant. The use of flumazenil mitigated the patient's IH symptoms including excessive daytime sleepiness, sleep drunkenness and self-reported cognitive problems. The case both provides a possible treatment and system for short (subcutaneous (SC) administration) and longer term (implant) flumazenil delivery. Current data supports the need for further research into the use of flumazenil for the treatment of IH and to develop long term flumazenil delivery systems.
authors.affiliation: Fresh Start Recovery Programme, Subiaco, WA, Australia School of Psychiatry and Clinical Neurosciences, University of Western Australia, Crawley WA, Australia erin.kelty@freshstart.org.au.
authors.forename: Erin
authors.initials: E
authors.lastname: Kelty
authors.affiliation: Fresh Start Recovery Programme, Subiaco, WA, Australia.
authors.forename: Vlad
authors.initials: V
authors.lastname: Martyn
authors.affiliation: Fresh Start Recovery Programme, Subiaco, WA, Australia.
authors.forename: George
authors.initials: G
authors.lastname: O'Neil
authors.affiliation: School of Psychiatry and Clinical Neurosciences, University of Western Australia, Crawley WA, Australia.
authors.forename: Gary
authors.initials: G
authors.lastname: Hulse
mesh_terms: D000328:Adult; D004343:Drug Implants; D005442:Flumazenil / Q000008:administration & dosage*; D018757:GABA Modulators / Q000008:administration & dosage*; D006801:Humans; D020177:Idiopathic Hypersomnia / Q000175:diagnosis / Q000188:drug therapy* / Q000503:physiopathology; D055104:Infusions, Subcutaneous; D008297:Male; D012890:Sleep / Q000187:drug effects*; D016896:Treatment Outcome; D014851:Wakefulness / Q000187:drug effects*; D064690:Wakefulness-Promoting Agents / Q000008:administration & dosage*
chemical_list: D004343:Drug Implants; D018757:GABA Modulators; D064690:Wakefulness-Promoting Agents; D005442:Flumazenil
keywords: Idiopathic hypersomnia; flumazenil; subcutaneous infusion