Pitolisant, an inverse agonist of the histamine H3 receptor: an alternative stimulant for narcolepsy-cataplexy in teenagers with refractory sleepiness

Clinical Neuropharmacology
Clara InocentePatricia Franco

Abstract

Narcolepsy is a rare disabling sleep disorder characterized by excessive daytime sleepiness and cataplexy (sudden loss of muscle tone). Drugs such as pitolisant, which block histamine H3 autoreceptors, constitute a newly identified class of stimulants because they increase brain histamine and enhance wakefulness in animal and human adult narcolepsy. We report our experience with the off-label use of pitolisant in 4 teenagers with narcolepsy/cataplexy with severe daytime sleepiness, refractory to available treatments (modafinil, methylphenidate, mazindol, sodium oxybate, and D-amphetamine). All teenagers developed their disease during childhood (11.3 ± 2.4 years; 50% boys) and were 17.3 ± 0.8 years old at the time of pitolisant therapy. Pitolisant treatment was increased from 10 to 30 mg (n = 1) and 40 mg (n = 3). The adapted Epworth Sleepiness Score decreased from 14.3 ± 1.1 to 9.5 ± 2.9 (P = 0.03) with pitolisant alone to 7 ± 3.4 when combined with mazindol (n = 1), methylphenidate (n = 1), or sodium oxybate plus modafinil (n = 1). Mean sleep onset latency increased from 31 ± 14 minutes to 36 ± 8 minutes (P = 0.21) on the maintenance of wakefulness test. The severity and frequency of cataplexy were slightly improved. Adverse e...Continue Reading

References

Dec 1, 1994·Sleep·W A Broughton, R J Broughton
Jul 1, 1994·Journal of the American Academy of Child and Adolescent Psychiatry·R E DahlL Trubnick
Nov 1, 2003·Sleep Medicine·C HedmanV V Myllylä
Nov 11, 2003·Sleep Medicine·Anna IvanenkoDavid Gozal
Dec 14, 2004·Sleep·Xianchen Liu
Oct 4, 2006·Pediatrics·Sanford M MelzerMark S Reuben
Mar 10, 2009·Sleep Medicine·Deborah L ZvosecBrad J Hall

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Citations

Oct 16, 2012·Neurotherapeutics : the Journal of the American Society for Experimental NeuroTherapeutics·Emmanuel J M Mignot
Sep 8, 2012·The Journal of Neuroscience : the Official Journal of the Society for Neuroscience·Christian R Burgess, Thomas E Scammell
Jan 11, 2014·Der Nervenarzt·G Mayer
Oct 15, 2013·Drugs·Alberto K De la Herrán-Arita, Fabio García-García
Nov 13, 2015·Expert Opinion on Emerging Drugs·Shannon S Sullivan, Christian Guilleminault
Dec 2, 2015·Neurodegenerative Disease Management·Daniel J van WamelenNasir A Aziz
Nov 27, 2014·Annals of General Psychiatry·Diego BaronioRudimar Riesgo
Oct 18, 2013·Expert Opinion on Therapeutic Patents·Dorota Łażewska, Katarzyna Kieć-Kononowicz
Jun 24, 2015·Nature Reviews. Neurology·Ling ShanJerome M Siegel
Jul 21, 2016·Drugs·Yahiya Y Syed
Oct 30, 2016·European Journal of Pharmacology·Christian BrabantEzio Tirelli
Oct 19, 2016·Molecular Pharmacology·Gustavo Nieto-AlamillaJosé-Antonio Arias-Montaño
Aug 8, 2014·Neuroscience and Biobehavioral Reviews·Erich H SchneiderRoland Seifert
Sep 19, 2018·CNS Neuroscience & Therapeutics·Patricia FrancoJian-Sheng Lin
Apr 12, 2013·CNS Neuroscience & Therapeutics·Clara Odilia InocentePatricia Franco
Sep 17, 2013·Annals of Neurology·Clifford B Saper, Thomas E Scammell
Apr 30, 2014·Behavioural Pharmacology·Bassem SadekKatarzyna Kieć-Kononowicz
Nov 10, 2013·The Journal of Pharmacology and Experimental Therapeutics·Andria L Del TrediciEthan S Burstein
Oct 9, 2013·The Journal of Pharmacology and Experimental Therapeutics·Detlef NeumannRoland Seifert
Aug 29, 2014·Current Opinion in Pulmonary Medicine·Panagis Drakatos, Guy D Leschziner
Jan 18, 2018·Expert Opinion on Therapeutic Patents·Dorota Łażewska, Katarzyna Kieć-Kononowicz
Dec 16, 2019·Acta Neurologica Scandinavica·Kristian BolinAnne-Marie Landtblom
Jan 16, 2020·Expert Review of Clinical Pharmacology·Shuang Li, Junyi Yang
Jul 11, 2019·Sleep & Breathing = Schlaf & Atmung·Christian FranceschiniGiuseppe Plazzi
Jul 12, 2020·Current Neurology and Neuroscience Reports·Arpita ParmarIndra Narang
Sep 18, 2020·Expert Opinion on Pharmacotherapy·Stefano de BiaseMariarosaria Valente
May 23, 2018·Proceedings of the National Academy of Sciences of the United States of America·Mahesh K KaushikMasashi Yanagisawa
Jul 29, 2020·Molecules : a Journal of Synthetic Chemistry and Natural Product Chemistry·Agnieszka Zagórska, Anna Jaromin
Aug 9, 2020·Nature and Science of Sleep·Emily C BarkerKatherine M Sharkey
Apr 23, 2021·Neurotherapeutics : the Journal of the American Society for Experimental NeuroTherapeutics·Christian FranceschiniGiuseppe Plazzi
Jan 27, 2017·Translational Neuroscience·Kanchan SharmaElizabeth Coulthard
May 27, 2021·The Journal of Pediatric Pharmacology and Therapeutics : JPPT : the Official Journal of PPAG·Stephanie PenningtonElise Sannar
Jun 27, 2021·Journal of Sleep Research·Claudio L A BassettiGert J Lammers
Jun 27, 2021·European Journal of Neurology : the Official Journal of the European Federation of Neurological Societies·Claudio L A BassettiGert J Lammers
Aug 3, 2021·Frontiers in Neuroscience·Elliott Carthy, Tommas Ellender

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