| Literature DB >> 20160349 |
Vesna Cvetkovic-Lopes1, Laurence Bayer, Stéphane Dorsaz, Stéphanie Maret, Sylvain Pradervand, Yves Dauvilliers, Michel Lecendreux, Gert-Jan Lammers, Claire E H M Donjacour, Renaud A Du Pasquier, Corinne Pfister, Brice Petit, Hyun Hor, Michel Mühlethaler, Mehdi Tafti.
Abstract
Narcolepsy is a sleep disorder characterized by excessive daytime sleepiness and attacks of muscle atonia triggered by strong emotions (cataplexy). Narcolepsy is caused by hypocretin (orexin) deficiency, paralleled by a dramatic loss in hypothalamic hypocretin-producing neurons. It is believed that narcolepsy is an autoimmune disorder, although definitive proof of this, such as the presence of autoantibodies, is still lacking. We engineered a transgenic mouse model to identify peptides enriched within hypocretin-producing neurons that could serve as potential autoimmune targets. Initial analysis indicated that the transcript encoding Tribbles homolog 2 (Trib2), previously identified as an autoantigen in autoimmune uveitis, was enriched in hypocretin neurons in these mice. ELISA analysis showed that sera from narcolepsy patients with cataplexy had higher Trib2-specific antibody titers compared with either normal controls or patients with idiopathic hypersomnia, multiple sclerosis, or other inflammatory neurological disorders. Trib2-specific antibody titers were highest early after narcolepsy onset, sharply decreased within 2-3 years, and then stabilized at levels substantially higher than that of controls for up to 30 years. High Trib2-specific antibody titers correlated with the severity of cataplexy. Serum of a patient showed specific immunoreactivity with over 86% of hypocretin neurons in the mouse hypothalamus. Thus, we have identified reactive autoantibodies in human narcolepsy, providing evidence that narcolepsy is an autoimmune disorder.Entities:
Mesh:
Substances:
Year: 2010 PMID: 20160349 PMCID: PMC2827962 DOI: 10.1172/JCI41366
Source DB: PubMed Journal: J Clin Invest ISSN: 0021-9738 Impact factor: 14.808