{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,3]],"date-time":"2025-09-03T12:21:11Z","timestamp":1756902071959,"version":"3.44.0"},"reference-count":29,"publisher":"The Pediatric Pharmacy Association","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021]]},"abstract":"This case report evaluates the potential benefit of pitolisant in a 15-year-old female with Prader-Willi syndrome, obsessive-compulsive disorder, autism spectrum disorder, and mild intellectual disability. Due to its action on the H3 receptor, it enhances central activity of histaminergic neurons resulting in increased alertness, irrespective of the loss of orexin neurons seen in narcolepsy. Additionally, it is thought to modulate various other neurotransmitter systems including acetylcholine, norepinephrine, and dopamine. Pitolisant has the potential to improve many symptoms in patients with Prader-Willi syndrome and it appears to be well tolerated with minimal side effects observed. Therefore, the use of pitolisant should be considered in patients with Prader-Willi syndrome who fail a psychostimulant trial.<\/jats:p>","DOI":"10.5863\/1551-6776-26.4.405","type":"journal-article","created":{"date-parts":[[2021,5,19]],"date-time":"2021-05-19T18:17:42Z","timestamp":1621448262000},"page":"405-410","source":"Crossref","is-referenced-by-count":6,"title":["Pitolisant in an Adolescent with Prader-Willi Syndrome"],"prefix":"10.5863","volume":"26","author":[{"given":"Stephanie","family":"Pennington","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Danielle","family":"Stutzman","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Elise","family":"Sannar","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"3883","reference":[{"issue":"3","key":"i1551-6776-26-4-405-b1","series-title":"CNS 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De Cock\n V,\n \n \n Diene\n G,\n \n \n Molinas\n C,\n \n \n .\n\t\t\t\t\tEfficacy of modafinil on excessive daytime sleepiness in Prader-Willi Syndrome.\n\t\t\t\t\tAm J Med Genet A.\n\t\t\t\t\t2011;\n\t\t\t\t\t155A(\n\t\t\t\t\t7):\n\t\t\t\t\t1552\u2013\n\t\t\t\t\t1557."},{"key":"i1551-6776-26-4-405-b8","unstructured":"Wakix (pitolisant) [package insert].\n\t\t\t\t\tProuvy, France; Rottendorf, France. http:\/\/www.ema.europa.eu","article-title":"Wakix (pitolisant) [package insert]"},{"issue":"2","key":"i1551-6776-26-4-405-b9","series-title":"Clin Neuropharmacol","first-page":"55","article-title":"Pitolisant, an inverse agonist of the histamine H3 receptor: an alternative stimulant for narcolepsy-cataplexy in teenagers with refractory sleepiness","volume":"35","year":"2012","unstructured":"Inocente\n C,\n \n \n Arnulf\n I,\n \n \n Bastuji\n H,\n \n \n .\n\t\t\t\t\tPitolisant, an inverse agonist of the histamine H3 receptor: an alternative stimulant for narcolepsy-cataplexy in teenagers 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drug-resistant idiopathic and symptomatic hypersomnia: a chart review","volume":"15","year":"2014","unstructured":"Leu-Semenescu\n S,\n \n \n Nittur\n N,\n \n \n Golmard\n JL,\n \n \n Arnulf\n I.\n \n \n Effects of pitolisant, a histamine H3 inverse agonist, in drug-resistant idiopathic and symptomatic hypersomnia: a chart review.\n\t\t\t\t\tSleep Med.\n\t\t\t\t\t2014;\n\t\t\t\t\t15(\n\t\t\t\t\t6):\n\t\t\t\t\t681\u2013\n\t\t\t\t\t687."},{"issue":"13","key":"i1551-6776-26-4-405-b12","series-title":"Drugs","first-page":"1313","article-title":"Pitolisant: first global approval","volume":"76","year":"2016","unstructured":"Syed\n Y.\n \n \n Pitolisant: first global approval.\n\t\t\t\t\tDrugs.\n\t\t\t\t\t2016;\n\t\t\t\t\t76(\n\t\t\t\t\t13):\n\t\t\t\t\t1313\u2013\n\t\t\t\t\t1318."},{"issue":"3","key":"i1551-6776-26-4-405-b13","series-title":"Lancet Neurol","first-page":"200","article-title":"Safety and efficacy of pitolisant on cataplexy in patients with narcolepsy: a randomised, double-blind, placebo-controlled trial","volume":"16","year":"2017","unstructured":"Szakacs\n Z,\n \n \n Dauvilliers\n Y,\n \n \n Mikhaylov\n V,\n \n \n .\n\t\t\t\t\tSafety and efficacy of pitolisant on cataplexy in patients with narcolepsy: a randomised, double-blind, placebo-controlled trial.\n\t\t\t\t\tLancet Neurol.\n\t\t\t\t\t2017;\n\t\t\t\t\t16(\n\t\t\t\t\t3):\n\t\t\t\t\t200\u2013\n\t\t\t\t\t207."},{"issue":"7","key":"i1551-6776-26-4-405-b14","series-title":"Br J Pharmacol","first-page":"1398","article-title":"Histamine H3 receptor antagonist in relation to epilepsy and neurodegeneration: a systemic consideration of recent progress and perspectives","volume":"167","year":"2012","unstructured":"Bhowmik\n M,\n \n \n Khanam\n R,\n \n \n Vohora\n D.\n \n \n Histamine H3 receptor antagonist in relation to epilepsy and neurodegeneration: a systemic consideration of recent progress and perspectives.\n\t\t\t\t\tBr J 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Pulmonol","first-page":"209","article-title":"Sleep and breathing in Prader-Willi syndrome","volume":"34","year":"2002","unstructured":"Nixon\n GM,\n \n \n Brouillette\n RT.\n \n \n Sleep and breathing in Prader-Willi syndrome.\n\t\t\t\t\tPediatr Pulmonol.\n\t\t\t\t\t2002;\n\t\t\t\t\t34(\n\t\t\t\t\t3):\n\t\t\t\t\t209\u2013\n\t\t\t\t\t217."},{"key":"i1551-6776-26-4-405-b21","series-title":"Nat Sci Sleep","first-page":"127","article-title":"Update on the treatment of narcolepsy: clinical efficacy of pitolisant","volume":"9","year":"2017","unstructured":"Calik\n MW.\n \n \n Update on the treatment of narcolepsy: clinical efficacy of pitolisant.\n\t\t\t\t\tNat Sci Sleep.\n\t\t\t\t\t2017;\n\t\t\t\t\t9:\n\t\t\t\t\t127\u2013\n\t\t\t\t\t133."},{"key":"i1551-6776-26-4-405-b22","series-title":"Sleep Med","first-page":"125","article-title":"The European Medicines Agency review of pitolisant for treatment of narcolepsy: summary of the scientific assessment by the Committee for Medicinal Products for Human Use","volume":"33","year":"2017","unstructured":"Kollb-Sielecka\n M,\n \n \n Demolis\n P,\n \n \n Emmerich\n J,\n \n \n .\n\t\t\t\t\tThe European Medicines Agency review of pitolisant for treatment of narcolepsy: summary of the scientific assessment by the Committee for Medicinal Products for Human Use.\n\t\t\t\t\tSleep Med.\n\t\t\t\t\t2017;\n\t\t\t\t\t33:\n\t\t\t\t\t125\u2013\n\t\t\t\t\t129."},{"key":"i1551-6776-26-4-405-b23","series-title":"Neuropharmacology","first-page":"85","article-title":"Histamine and histamine receptors in Tourette syndrome and other neuropsychiatric conditions","volume":"106","year":"2016","unstructured":"Rapanelli\n M,\n \n \n Pittenger\n C.\n \n \n Histamine and histamine receptors in Tourette syndrome and other neuropsychiatric conditions.\n\t\t\t\t\tNeuropharmacology.\n\t\t\t\t\t2016;\n\t\t\t\t\t106:\n\t\t\t\t\t85\u2013\n\t\t\t\t\t90."},{"issue":"2","key":"i1551-6776-26-4-405-b24","series-title":"J Pediatr Pharmacol Ther","first-page":"166","article-title":"Cognitive improvements in children with Prader-Willi syndrome following pitolisant treatment \u2013 patient reports","volume":"24","year":"2019","unstructured":"Pullen\n L,\n \n \n Picone\n M,\n \n \n Tan\n L,\n \n \n .\n\t\t\t\t\tCognitive improvements in children with Prader-Willi syndrome following pitolisant treatment \u2013 patient reports.\n\t\t\t\t\tJ Pediatr Pharmacol Ther.\n\t\t\t\t\t2019;\n\t\t\t\t\t24(\n\t\t\t\t\t2):\n\t\t\t\t\t166\u2013\n\t\t\t\t\t171."},{"issue":"11","key":"i1551-6776-26-4-405-b25","series-title":"Lancet Neurol","first-page":"1068","article-title":"Pitolisant versus placebo or modafinil in patients with narcolepsy: a double-blind, randomised trial","volume":"12","year":"2013","unstructured":"Dauvilliers\n Y,\n \n \n Bassetti\n C,\n \n \n Lammers\n GJ,\n \n \n .\n\t\t\t\t\tPitolisant versus placebo or modafinil in patients with narcolepsy: a double-blind, randomised trial.\n\t\t\t\t\tLancet Neurol.\n\t\t\t\t\t2013;\n\t\t\t\t\t12(\n\t\t\t\t\t11):\n\t\t\t\t\t1068\u2013\n\t\t\t\t\t1075."},{"key":"i1551-6776-26-4-405-b26","unstructured":"Chion Foundation.\n Chion Foundation. Pitolisant Program USA. Accessed June 26, 2018. https:\/\/www.chionfoundation.org\/pitolisant-usa"},{"key":"i1551-6776-26-4-405-b27","unstructured":"Chion Foundation. The Chion Foundation and TREND Community Partner to Identify Novel Treatment for Prader-Willi Syndrome.\n\t\t\t\t\tAccessed June 26, 2018. https:\/\/www.chionfoundation.org\/single-post\/2017\/10\/06\/press-release-the-chion-foundation-and-trend-community-partner-to-identify-novel-treatmen","article-title":"Chion Foundation. The Chion Foundation and TREND Community Partner to Identify Novel Treatment for Prader-Willi Syndrome"},{"issue":"11","key":"i1551-6776-26-4-405-b28","series-title":"Expert Opin Pharmacother","first-page":"1615","article-title":"Pharmacotherapy to control behavioral symptoms in children with autism","volume":"13","year":"2012","unstructured":"Doyle\n CA,\n \n \n McDougle\n CJ.\n \n \n Pharmacotherapy to control behavioral symptoms in children with autism.\n\t\t\t\t\tExpert Opin Pharmacother.\n\t\t\t\t\t2012;\n\t\t\t\t\t13(\n\t\t\t\t\t11):\n\t\t\t\t\t1615\u2013\n\t\t\t\t\t1629."},{"issue":"10","key":"i1551-6776-26-4-405-b29","series-title":"JAMA Psychiatry","first-page":"1067","article-title":"Antipsychotics and the risk of type 2 diabetes mellitus in children and youth","volume":"70","year":"2013","unstructured":"Bobo\n WV,\n \n \n Cooper\n WO,\n \n \n Stein\n CM,\n \n \n .\n\t\t\t\t\tAntipsychotics and the risk of type 2 diabetes mellitus in children and youth.\n\t\t\t\t\tJAMA Psychiatry.\n\t\t\t\t\t2013;\n\t\t\t\t\t70(\n\t\t\t\t\t10):\n\t\t\t\t\t1067\u2013\n\t\t\t\t\t1075."}],"container-title":["The Journal of Pediatric Pharmacology and Therapeutics"],"original-title":[],"link":[{"URL":"https:\/\/jppt.kglmeridian.com\/view\/journals\/jppt\/26\/4\/article-p405.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/meridian.allenpress.com\/jppt\/article-pdf\/26\/4\/405\/2830035\/i1551-6776-26-4-405.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/jppt.kglmeridian.com\/downloadpdf\/journals\/jppt\/26\/4\/article-p405.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/jppt.kglmeridian.com\/downloadpdf\/journals\/jppt\/26\/4\/article-p405.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,5,16]],"date-time":"2025-05-16T00:44:49Z","timestamp":1747356289000},"score":1,"resource":{"primary":{"URL":"https:\/\/jppt.kglmeridian.com\/view\/journals\/jppt\/26\/4\/article-p405.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021]]},"references-count":29,"journal-issue":{"issue":"4"},"URL":"https:\/\/doi.org\/10.5863\/1551-6776-26.4.405","relation":{},"ISSN":["1551-6776"],"issn-type":[{"type":"print","value":"1551-6776"}],"subject":[],"published":{"date-parts":[[2021]]}}}