{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T02:03:17Z","timestamp":1791511397312,"version":"4.3.4"},"reference-count":31,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2021,6,17]],"date-time":"2021-06-17T00:00:00Z","timestamp":1623888000000},"content-version":"vor","delay-in-days":77,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"The National Reference Center for Kleine-Levin syndrome"},{"name":"French Health Ministry"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,4,5]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Kleine\u2013Levin syndrome is a rare disorder characterized by relapsing-remitting episodes of severe hypersomnia, cognitive impairment, apathy, derealization and behavioural disturbances. Between episodes, most patients experience normal sleep, mood and behaviour, but they may have some residual abnormalities in brain functional imaging; however, the frequency, localization and significance of abnormal imaging are unknown, as brain functional imaging have been scarce and heterogenous [including scintigraphy 18F-fluorodeoxyglucose positron emission tomography\/computerized tomography (FDG-PET\/CT) and functional MRI during resting state and cognitive effort] and based on case reports or on group analysis in small groups. Using visual individual analysis of 18F-fluorodeoxyglucose positron emission tomography\/computerized tomography at the time of Kleine\u2013Levin syndrome diagnosis, we examined the frequency, localization and clinical determinants of hypo- and hypermetabolism in a cross-sectional study. Among 179 patients with Kleine\u2013Levin syndrome who underwent 18F-fluorodeoxyglucose positron emission tomography\/computerized tomography, the visual analysis was restricted to the 138 untreated patients studied during asymptomatic periods. As many as 70% of patients had hypometabolism, mostly affecting the posterior associative cortex and the hippocampus. Hypometabolism was associated with younger age, recent (&amp;lt;3\u2009years) disease course and a higher number of episodes during the preceding year. The hypometabolism was more extensive (from the left temporo-occipital junction to the entire homolateral and then the bilateral posterior associative cortex) at the beginning of the disorder. In contrast, there was hypermetabolism in the prefrontal dorsolateral cortex in half of the patients (almost all having concomitant hypometabolism in the posterior areas), which was also associated with younger age and shorter disease course. The cognitive performances (including episodic memory) were similar in patients with versus without hippocampus hypometabolism. In conclusion, hypometabolism is frequently observed upon individual visual analysis of 18F-fluorodeoxyglucose positron emission tomography\/computerized tomography during asymptomatic Kleine\u2013Levin syndrome periods; it is mostly affecting the posterior associative cortex and the hippocampus and is mostly in young patients with recent-onset disease. Hypometabolism provides a trait marker during the first years of Kleine\u2013Levin syndrome, which could help clinicians during the diagnosis process.<\/jats:p>","DOI":"10.1093\/braincomms\/fcab130","type":"journal-article","created":{"date-parts":[[2021,6,16]],"date-time":"2021-06-16T07:12:39Z","timestamp":1623827559000},"source":"Crossref","is-referenced-by-count":11,"title":["Functional brain imaging using 18F-fluorodeoxyglucose positron emission tomography\/computerized tomography in 138 patients with Kleine\u2013Levin syndrome: an early marker?"],"prefix":"10.1093","volume":"3","author":[{"given":"Benjamin","family":"Dudoignon","sequence":"first","affiliation":[{"name":"Sorbonne University, Paris Brain Institute (ICM), Inserm UMR-S975, CNRS UMR7225, Paris 75013, France"},{"name":"Sleep Disorders Unit, National Reference Centre for Kleine-Levin Syndrome, Piti\u00e9 Salp\u00eatri\u00e8re University Hospital, APHP, Paris 75013, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laure-Eug\u00e9nie","family":"Tainturier","sequence":"additional","affiliation":[{"name":"Nuclear Medicine Department, H\u00f4pitaux Universitaires Piti\u00e9-Salp\u00eatri\u00e8re University Hospital, Sorbonne University, AP-HP, Paris 75013, France"},{"name":"LIB, INSERM U1146, 75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pauline","family":"Dodet","sequence":"additional","affiliation":[{"name":"Sorbonne University, Paris Brain Institute (ICM), Inserm UMR-S975, CNRS UMR7225, Paris 75013, France"},{"name":"Sleep Disorders Unit, National Reference Centre for Kleine-Levin Syndrome, Piti\u00e9 Salp\u00eatri\u00e8re University Hospital, APHP, Paris 75013, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G\u00e9raldine","family":"Bera","sequence":"additional","affiliation":[{"name":"Nuclear Medicine Department, H\u00f4pitaux Universitaires Piti\u00e9-Salp\u00eatri\u00e8re University Hospital, Sorbonne University, AP-HP, Paris 75013, France"},{"name":"LIB, INSERM U1146, 75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisabeth","family":"Groos","sequence":"additional","affiliation":[{"name":"Sorbonne University, Paris Brain Institute (ICM), Inserm UMR-S975, CNRS UMR7225, Paris 75013, France"},{"name":"Sleep Disorders Unit, National Reference Centre for Kleine-Levin Syndrome, Piti\u00e9 Salp\u00eatri\u00e8re University Hospital, APHP, Paris 75013, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charlotte","family":"Chaumereuil","sequence":"additional","affiliation":[{"name":"Sorbonne University, Paris Brain Institute (ICM), Inserm UMR-S975, CNRS UMR7225, Paris 75013, France"},{"name":"Sleep Disorders Unit, National Reference Centre for Kleine-Levin Syndrome, Piti\u00e9 Salp\u00eatri\u00e8re University Hospital, APHP, Paris 75013, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean-Baptiste","family":"Maranci","sequence":"additional","affiliation":[{"name":"Sorbonne University, Paris Brain Institute (ICM), Inserm UMR-S975, CNRS UMR7225, Paris 75013, France"},{"name":"Sleep Disorders Unit, National Reference Centre for Kleine-Levin Syndrome, Piti\u00e9 Salp\u00eatri\u00e8re University Hospital, APHP, Paris 75013, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aur\u00e9lie","family":"Kas","sequence":"additional","affiliation":[{"name":"Nuclear Medicine Department, H\u00f4pitaux Universitaires Piti\u00e9-Salp\u00eatri\u00e8re University Hospital, Sorbonne University, AP-HP, Paris 75013, France"},{"name":"LIB, INSERM U1146, 75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2240-2516","authenticated-orcid":false,"given":"Isabelle","family":"Arnulf","sequence":"additional","affiliation":[{"name":"Sorbonne University, Paris Brain Institute (ICM), Inserm UMR-S975, CNRS UMR7225, Paris 75013, France"},{"name":"Sleep Disorders Unit, National Reference Centre for Kleine-Levin Syndrome, Piti\u00e9 Salp\u00eatri\u00e8re University Hospital, APHP, Paris 75013, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2021,6,17]]},"reference":[{"issue":"10","key":"2021121218085408500_fcab130-B1","doi-asserted-by":"crossref","first-page":"918","DOI":"10.1016\/S1474-4422(12)70187-4","article-title":"Diagnosis, disease course, and management of patients with Kleine-Levin syndrome","volume":"11","author":"Arnulf","year":"2012","journal-title":"Lancet 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