Elevated Tribbles homolog 2-specific antibody levels in narcolepsy patients.
| Data(s) |
2010
|
|---|---|
| Resumo |
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. |
| Identificador |
https://serval.unil.ch/?id=serval:BIB_6F1CF4DFB5CB isbn:1558-8238[electronic], 0021-9738[linking] pmid:20160349 doi:10.1172/JCI41366 isiid:000275272700010 |
| Idioma(s) |
en |
| Fonte |
Journal of Clinical Investigation, vol. 120, no. 3, pp. 713-719 |
| Palavras-Chave | #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 |
| Tipo |
info:eu-repo/semantics/article article |