{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T19:03:05Z","timestamp":1792782185929,"version":"4.5.0"},"reference-count":32,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2016,1,6]],"date-time":"2016-01-06T00:00:00Z","timestamp":1452038400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports and Technology (MEXT)","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Ministry of Health, Labour and Welfare of Japan","id":[{"id":"https:\/\/ror.org\/03mwa7s65","id-type":"ROR","asserted-by":"crossref"}]},{"DOI":"10.13039\/100012045","name":"Kawano Masanori Memorial Public Interest Incorporated Foundation for Promotion of Pediatrics","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100012045","id-type":"DOI","asserted-by":"crossref"}]},{"name":"International Institute for Integrative Sleep Medicine (IIIS), sponsored by the World Premier International Research Center Initiative (WPI), from MEXT, Japan","id":[{"id":"https:\/\/ror.org\/01s1qrh29","id-type":"ROR","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2016,5]]},"abstract":"<jats:sec>\n                    <jats:label\/>\n                    <jats:p>Prader\u2013Willi syndrome is a congenital neurodevelopmental disorder resulting from deletion of the paternal copies of genes within the chromosome region 15q11\u2010q13. Patients with Prader\u2013Willi syndrome often exhibit excessive daytime sleepiness, excessive appetite, and obesity. As is the case in narcolepsy, orexin (hypocretin) may be responsible for these symptoms. However, reports showing cerebrospinal fluid orexin levels in Prader\u2013Willi syndrome patients have been limited. The aim of this study was to examine the relationship between the characteristic symptoms of Prader\u2013Willi syndrome and cerebrospinal fluid orexin levels. We clinically identified 14 Prader\u2013Willi syndrome patients and examined their cerebrospinal fluid orexin levels. A total of 12 patients with a 15q11\u2010q13 deletion and two patients with maternal uniparental disomy of chromosome 15 were identified. A total of 37 narcoleptic patients and 14 idiopathic hypersomnia patients were recruited for comparison. Cerebrospinal fluid orexin levels (median [25\u201375 percentiles]) in the 14 Prader\u2013Willi syndrome patients were intermediate (192 [161\u2013234.5]\u2009pg\/ml), higher than in the narcoleptic patients, but lower than in the idiopathic hypersomnia patients. Body mass index of the Prader\u2013Willi syndrome patients was higher than in the narcoleptic and idiopathic hypersomnia patients. There was also a negative correlation between Epworth sleepiness scale scores and orexin levels in Prader\u2013Willi syndrome patients. Decreased cerebrospinal fluid orexin levels in Prader\u2013Willi syndrome may play an important role in severity of obesity and excessive daytime sleepiness. \u00a9 2016 Wiley Periodicals, Inc.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ajmg.a.37542","type":"journal-article","created":{"date-parts":[[2016,1,7]],"date-time":"2016-01-07T07:24:01Z","timestamp":1452151441000},"page":"1181-1186","source":"Crossref","is-referenced-by-count":42,"title":["Decline of CSF orexin (hypocretin) levels in Prader\u2013Willi syndrome"],"prefix":"10.1002","volume":"170","author":[{"given":"Mayu","family":"Omokawa","sequence":"first","affiliation":[{"name":"Department of Neuropsychiatry Akita University Graduate School of Medicine Akita Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tadayuki","family":"Ayabe","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Koshigaya 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