Positive oligoclonal bands and CSF pleocytosis in narcolepsy type 1: A case report supporting the immune-mediated hypothesis.

作者: Ana Moreno-Estébanez , Iker Bilbao Villabeitia , Teresa Echeverria Guibert , Mar Mendibe Bilbao , Sabas Boyero Durán

DOI: 10.1016/J.JNEUROIM.2019.577111

关键词: CSF pleocytosisPleocytosisAutoimmune encephalitisMedicineExcessive daytime sleepinessCataplexyOrexinNarcolepsyHypnopompicImmunology

摘要: Abstract Narcolepsy-type 1 is a neurological sleep-disorder caused by selective loss of hypothalamic orexin/hypocretin-producing neurons whose underlying mechanism considered to be immune-mediated. We report the case 16 year-old girl with excessive daytime sleepiness, hypnagogic/hypnopompic hallucinations and cataplexy, fulfilling narcolepsy-type diagnostic criteria. She was HLA-DQB1*06:02/DQA1*01:02 positive. CSF analysis demonstrated positive IgG oligoclonal bands, pleocytosis hypocretin-1 below detection limit. Other autoimmune processes were excluded, including encephalitis. After treatment intravenous immunoglobulins sleep-related transiently improved for month. This case's inflammatory findings support role neuroinflammation in development genetically predisposed patients.

参考文章(32)
J. D. Parkes, K. Welsh, N. Langdon, C. Lock, Immune Factors in Narcolepsy in the United Kingdom Springer, Berlin, Heidelberg. pp. 158- 171 ,(1988) , 10.1007/978-3-642-83387-8_14
K. Matsuki, T. Juji, Y. Honda, Immunological Features of Narcolepsy in Japan Springer, Berlin, Heidelberg. pp. 150- 157 ,(1988) , 10.1007/978-3-642-83387-8_13
E. Mignot, X. Lin, J. Arrigoni, C. Macaubas, F. Olive, J. Hallmayer, P. Underhill, C. Guilleminault, W. C. Dement, F. C. Grumet, DQB1*0602 and DQA1*0102 (DQ1) Are Better Markers Than DR2 for Narcolepsy in Caucasian and Black Americans Sleep. ,vol. 17, pp. S60- S67 ,(1994) , 10.1093/SLEEP/17.SUPPL_8.S60
Christelle Peyron, Juliette Faraco, William Rogers, Beth Ripley, Sebastiaan Overeem, Yves Charnay, Sona Nevsimalova, Michael Aldrich, David Reynolds, Roger Albin, Robin Li, Marcel Hungs, Mario Pedrazzoli, Muralidhara Padigaru, Melanie Kucherlapati, Jun Fan, Richard Maki, Gert Jan Lammers, Constantin Bouras, Raju Kucherlapati, Seiji Nishino, Emmanuel Mignot, None, A mutation in a case of early onset narcolepsy and a generalized absence of hypocretin peptides in human narcoleptic brains Nature Medicine. ,vol. 6, pp. 991- 997 ,(2000) , 10.1038/79690
Thomas C. Thannickal, Robert Nienhuis, Jerome M. Siegel, Localized loss of hypocretin (orexin) cells in narcolepsy without cataplexy Sleep. ,vol. 32, pp. 993- 998 ,(2009) , 10.1093/SLEEP/32.8.993
, Mehdi Tafti, Hyun Hor, Yves Dauvilliers, Gert J. Lammers, Sebastiaan Overeem, Geert Mayer, Sirous Javidi, Alex Iranzo, Joan Santamaria, Rosa Peraita-Adrados, José L. Vicario, Isabelle Arnulf, Giuseppe Plazzi, Sophie Bayard, Francesca Poli, Fabio Pizza, Peter Geisler, Aleksandra Wierzbicka, Claudio L. Bassetti, Johannes Mathis, Michel Lecendreux, Claire E.H.M. Donjacour, Astrid van der Heide, Raphaël Heinzer, José Haba-Rubio, Eva Feketeova, Birgit Högl, Birgit Frauscher, Antonio Benetó, Ramin Khatami, Francesca Cañellas, Corinne Pfister, Sabine Scholz, Michel Billiard, Christian R. Baumann, Guadalupe Ercilla, Willem Verduijn, Frans H.J. Claas, Valérie Dubois, Jacek Nowak, Hans-Peter Eberhard, Sylvain Pradervand, Charlotte N. Hor, Manuela Testi, Jean-Marie Tiercy, Zoltán Kutalik, DQB1 locus alone explains most of the risk and protection in narcolepsy with cataplexy in Europe. Sleep. ,vol. 37, pp. 19- 25 ,(2014) , 10.5665/SLEEP.3300
S. Fredrikson, B. Carlander, M. Billiard, H. Link, CSF immune variables in patients with narcolepsy. Acta Neurologica Scandinavica. ,vol. 81, pp. 253- 254 ,(2009) , 10.1111/J.1600-0404.1990.TB00978.X
Thomas C. Thannickal, Robert Y. Moore, Robert Nienhuis, Lalini Ramanathan, Seema Gulyani, Michael Aldrich, Marsha Cornford, Jerome M. Siegel, Reduced number of hypocretin neurons in human narcolepsy. Neuron. ,vol. 27, pp. 469- 474 ,(2000) , 10.1016/S0896-6273(00)00058-1
P. O. Valko, R. Khatami, C. R. Baumann, C. L. Bassetti, No persistent effect of intravenous immunoglobulins in patients with narcolepsy with cataplexy. Journal of Neurology. ,vol. 255, pp. 1900- 1903 ,(2008) , 10.1007/S00415-008-0996-2
Yves Dauvilliers, Bertrand Carlander, François, Jacques Touchon, Mehdi Tafti, Successful management of cataplexy with intravenous immunoglobulins at narcolepsy onset. Annals of Neurology. ,vol. 56, pp. 905- 908 ,(2004) , 10.1002/ANA.20339