Elevated Tribbles homolog 2-specific antibody levels in narcolepsy patients.

Details

Serval ID
serval:BIB_6F1CF4DFB5CB
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
Elevated Tribbles homolog 2-specific antibody levels in narcolepsy patients.
Journal
Journal of Clinical Investigation
Author(s)
Cvetkovic-Lopes V., Bayer L., Dorsaz S., Maret S., Pradervand S., Dauvilliers Y., Lecendreux M., Lammers G.J., Donjacour C.E., Du Pasquier R.A., Pfister C., Petit B., Hor H., Mühlethaler M., Tafti M.
ISSN
1558-8238[electronic], 0021-9738[linking]
Publication state
Published
Issued date
2010
Peer-reviewed
Oui
Volume
120
Number
3
Pages
713-719
Language
english
Notes
Publication types: Journal Article ; Research Support, Non-U.S. Gov't Publication Status: ppublish
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.
Keywords
Animals, Autoantibodies/blood, Autoantibodies/immunology, Autoantigens/genetics, Autoantigens/immunology, Autoimmune Diseases/blood, Autoimmune Diseases/genetics, Female, Humans, Hypothalamus/immunology, Hypothalamus/metabolism, Intracellular Signaling Peptides and Proteins/genetics, Intracellular Signaling Peptides and Proteins/immunology, Male, Mice, Mice, Transgenic, Narcolepsy/blood, Narcolepsy/genetics, Neurons/immunology, Neurons/metabolism, Neuropeptides/genetics, Neuropeptides/immunology, Protein-Serine-Threonine Kinases/genetics, Protein-Serine-Threonine Kinases/immunology, Severity of Illness Index, Sleep Initiation and Maintenance Disorders/blood, Sleep Initiation and Maintenance Disorders/genetics, Time Factors
Pubmed
Web of science
Open Access
Yes
Create date
24/01/2011 13:42
Last modification date
20/08/2019 15:28
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