Elevated Tribbles homolog 2-specific antibody levels in narcolepsy patients.
نویسندگان
چکیده
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.
منابع مشابه
NARCOLEPSY IS TIGHTLY ASSOCIATED WITH THE HUMAN LEUKOCYTE ANTIGEN (HLA) CLASS II RE- GION, WITH 95% OF NARCOLEPSY PATIENTS WITH cataplexy
869 Anti-Tribbles Homolog 2 Autoantibodies in Narcolepsy— Kawashima et al NARCOLEPSY IS TIGHTLY ASSOCIATED WITH THE HUMAN LEUKOCYTE ANTIGEN (HLA) CLASS II REGION, WITH 95% OF NARCOLEPSY PATIENTS WITH cataplexy carrying the HLA-DQB1*0602 allele.1 In most human narcolepsy cases with cataplexy, hypocretin-1 concentration is reduced or undetectable in the cerebrospinal fluid (CSF).2,3 The lack of C...
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ورودعنوان ژورنال:
- The Journal of clinical investigation
دوره 120 3 شماره
صفحات -
تاریخ انتشار 2010