| Literature DB >> 28202777 |
Tory P Johnson1, Richa Tyagi1, Paul R Lee1, Myoung-Hwa Lee1, Kory R Johnson2, Jeffrey Kowalak3, Abdel Elkahloun4, Marie Medynets5, Alina Hategan1, Joseph Kubofcik6, James Sejvar7, Jeffrey Ratto8, Sudhir Bunga8, Issa Makumbi9, Jane R Aceng9, Thomas B Nutman6, Scott F Dowell10, Avindra Nath11.
Abstract
Nodding syndrome is an epileptic disorder of unknown etiology that occurs in children in East Africa. There is an epidemiological association with Onchocerca volvulus, the parasitic worm that causes onchocerciasis (river blindness), but there is limited evidence that the parasite itself is neuroinvasive. We hypothesized that nodding syndrome may be an autoimmune-mediated disease. Using protein chip methodology, we detected autoantibodies to leiomodin-1 more abundantly in patients with nodding syndrome compared to unaffected controls from the same village. Leiomodin-1 autoantibodies were found in both the sera and cerebrospinal fluid of patients with nodding syndrome. Leiomodin-1 was found to be expressed in mature and developing human neurons in vitro and was localized in mouse brain to the CA3 region of the hippocampus, Purkinje cells in the cerebellum, and cortical neurons, structures that also appear to be affected in patients with nodding syndrome. Antibodies targeting leiomodin-1 were neurotoxic in vitro, and leiomodin-1 antibodies purified from patients with nodding syndrome were cross-reactive with O. volvulus antigens. This study provides initial evidence supporting the hypothesis that nodding syndrome is an autoimmune epileptic disorder caused by molecular mimicry with O. volvulus antigens and suggests that patients may benefit from immunomodulatory therapies.Entities:
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Year: 2017 PMID: 28202777 PMCID: PMC5434766 DOI: 10.1126/scitranslmed.aaf6953
Source DB: PubMed Journal: Sci Transl Med ISSN: 1946-6234 Impact factor: 17.956