{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,27]],"date-time":"2026-09-27T08:53:40Z","timestamp":1790499220853,"version":"4.1.0"},"reference-count":26,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2016,7,14]],"date-time":"2016-07-14T00:00:00Z","timestamp":1468454400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"French Ministry of Research and Higher Education"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Annals of Neurology"],"published-print":{"date-parts":[[2016,8]]},"abstract":"<jats:sec><jats:title>Objective<\/jats:title><jats:p>The pathophysiology of idiopathic hypersomnia (IH) remains unclear. Recently, cerebrospinal fluid (CSF)\u2010induced enhancement of \u03b3\u2010aminobutyric acid (GABA)\u2010A receptor activity was found in patients with IH compared to controls.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Fifteen unrelated patients (2 males and 13 females) affected with typical IH, 12 patients (9 males and 3 females) with narcolepsy type 1, and 15 controls (9 males and 6 females) with unspecified hypersomnolence (n\u2009=\u20097) and miscellaneous neurological conditions (n\u2009=\u20098) were included. A lumbar puncture was performed in all participants to measure CSF hypocretin\u20101 and GABA\u2010A response. We used a voltage\u2010clamp assay on <jats:italic>Xenopus<\/jats:italic> oocytes injected with the RNAs that encode the \u03b1<jats:sub>1<\/jats:sub>\u03b2<jats:sub>2<\/jats:sub>\u03b3<jats:sub>2<\/jats:sub> or the \u03b1<jats:sub>2<\/jats:sub>\u03b2<jats:sub>2<\/jats:sub>\u03b3<jats:sub>2<\/jats:sub> subunits of the human GABA\u2010A receptor. A sequence of 6 different applications (GABA, GABA\/CSF, and CSF alone) with 2 to 4 oocytes per CSF sample was performed in a whole\u2010cell voltage\u2010clamp assay.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Representative current traces from oocytes expressing human \u03b1<jats:sub>1<\/jats:sub>\u03b2<jats:sub>2<\/jats:sub>\u03b3<jats:sub>2<\/jats:sub> or \u03b1<jats:sub>2<\/jats:sub>\u03b2<jats:sub>2<\/jats:sub>\u03b3<jats:sub>2<\/jats:sub> GABA\u2010A receptors were recorded in response to 6 successive puffs of GABA diluted in the survival medium (SM), showing stable and reliable response. GABA puffs diluted in SM\/CSF solution or SM\/CSF solution alone showed no significant differences in the CSF of IH, narcolepsy, or control groups. No associations were found between GABA responses, demographic features, disease duration, or disease severity in the whole population or within groups.<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>Using the <jats:italic>Xenopus<\/jats:italic> oocyte assay, we found an absence of GABA\u2010A receptor potentiation with CSF from patients with central hypersomnolence disorders, with no significant differences between hypocretin\u2010deficient and non\u2013hypocretin\u2010deficient patients compared to controls. 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