ArticleEpilepsia open2021
Systemic and cerebrospinal fluid immune and complement activation in Ugandan children and adolescents with long-standing nodding syndrome: A case-control study.
Article in Epilepsia open, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.
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Who cites it
11 citing papers in PubMed, 16 citations in OpenAlex.
- Doxycycline for the treatment of nodding syndrome: a randomised, placebo-controlled, phase 2 trial.The Lancet. Global health · 2024Trial
- The Nodding syndrome cerebrospinal fluid proteome: a lens into neurodevelopmental failure consistent with environmentally triggeredFrontiers in molecular neuroscience · 2026Article
- Investigating HLA haplotypes as a potential risk factor for nodding syndrome: A case-control study in the Mahenge area, Tanzania.PLoS neglected tropical diseases · 2025Article
- A retrospective analysis ofAntimicrobial agents and chemotherapy · 2024Article
- Epilepsy and nodding syndrome in association with an Onchocerca volvulus infection drive distinct immune profile patterns.PLoS neglected tropical diseases · 2023Article
- Neuroimmunology of Common Parasitic Infections in Africa.Frontiers in immunology · 2022Review
- Nodding Syndrome: A Scoping Review.Tropical medicine and infectious disease · 2021Article
- Risk Factors for Nodding Syndrome and Other Forms of Epilepsy in Northern Uganda: A Case-Control Study.Pathogens (Basel, Switzerland) · 2021Article
- Systemic and cerebrospinal fluid immune and complement activation in Ugandan children and adolescents with long-standing nodding syndrome: A case-control study.Epilepsia open · 2021Article
- Cytokines and Onchocerciasis-Associated Epilepsy, a Pilot Study and Review of the Literature.Pathogens (Basel, Switzerland) · 2021Article
- Epilepsy inFrontiers in neurology · 2021Article
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Authors and funding
12 authors at 7 institutions in 4 countries.
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Abstract
objectiveNodding syndrome is a poorly understood epileptic encephalopathy characterized by a unique seizure type-head nodding-and associated with Onchocerca volvulus infection. We hypothesized that altered immune activation in the cerebrospinal fluid (CSF) and plasma of children with nodding syndrome may yield insights into the pathophysiology and progression of this seizure disorder.
methodWe conducted a case-control study of 154 children (8 years or older) with long-standing nodding syndrome and 154 healthy age-matched community controls in 3 districts of northern Uganda affected by nodding syndrome. Control CSF samples were obtained from Ugandan children in remission from hematological malignancy during routine follow-up. Markers of immune activation and inflammation (cytokines and chemokines) and complement activation (C5a) were measured in plasma and CSF using ELISA or Multiplex Luminex assays. O volvulus infection was assessed by serology for anti-OV-16 IgG levels.
resultsThe mean (SD) age of the population was 15.1 (SD: 1.9) years, and the mean duration of nodding syndrome from diagnosis to enrollment was 8.3 (SD: 2.7) years. The majority with nodding syndrome had been exposed to O volvulus (147/154 (95.4%)) compared with community children (86/154 (55.8%)), with an OR of 17.04 (95% CI: 7.33, 45.58), P < .001. C5a was elevated in CSF of children with nodding syndrome compared to controls (P < .0001). The levels of other CSF markers tested were comparable between cases and controls after adjusting for multiple comparisons. Children with nodding syndrome had lower plasma levels of IL-10, APRIL, CCL5 (RANTES), CCL2, CXCL13, and MMP-9 compared with community controls (P < .05 for all; multiple comparisons). Plasma CRP was elevated in children with nodding syndrome compared to community children and correlated with disease severity. SIGNIFICANCE: Nodding syndrome is associated with exposure to O. volvulus. Compared to controls, children with long-standing symptoms of nodding syndrome show evidence of complement activation in CSF and altered immune activation in plasma.
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