Preliminary seroprevalence study of neurotropic virus antibodies in Nodding syndrome

Angues, Raquel Valdes; Palmer, Valerie S.; Mazumder, Rajarshi; Okot, Caesar; Spencer, Peter S.

doi:10.1016/j.ensci.2022.100423

Cited by 4 publications

(3 citation statements)

References 21 publications

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“…NS has previously been associated with Onchocerca volvulus infection, 5–9 history of measles 10 , 11 and nutritional deficiencies (specifically vitamin B 6 deficiency), 8 , 12–14 which resulted in potentially premature public health interventions as proof of causality is lacking. 2 , 10 , 15 Part of the failure to identify the aetiology may be explained by the fact that previous studies were limited in sample size, diagnostic breadth and investigations during late-stage disease when a causative factor may no longer be detectable.…”

Section: Introductionmentioning

confidence: 99%

Parasitic, bacterial, viral, immune-mediated, metabolic and nutritional factors associated with nodding syndrome

Edridge,

Abd-Elfarag,

Deijs

et al. 2023

Brain Communications

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Nodding syndrome is a neglected, disabling and potentially fatal epileptic disorder of unknown aetiology affecting thousands of individuals mostly confined to Eastern sub-Saharan Africa. Previous studies have identified multiple associations – including O. volvulus, antileiomodin-1 antibodies, vitamin B6 deficiency, and measles virus infection – yet none is proven causal. We conducted a case-control study of children with early-stage Nodding syndrome (symptom onset <1 year). Cases and controls were identified through a household survey in the Greater Mundri area in South Sudan. A wide range of parasitic, bacterial, viral, immune-mediated, metabolic, and nutritional risk factors was investigated using conventional and state-of-the-art untargeted assays. Associations were examined by multiple logistic regression analysis and a hypothetical causal model was constructed using structural equation modelling. From 607 children with Nodding syndrome, 72 with early-stage disease were included as cases and matched to 65 household- and 44 community controls. Mansonella perstans infection (odds ratio [OR] 7.04, 95% confidence interval [CI] 2.28-21.7), Necator americanus infection (OR 2.33, 95% CI 1.02-5.3), higher antimalarial seroreactivity (OR 1.75, 95% CI 1.20-2.57), higher vitamin E concentration (OR 1.53 per standard deviation [SD] increase, 95% CI 1.07-2.19) and lower vitamin B12 concentration (OR 0.56 per SD increase, 95% CI 0.36-0.87) were associated with higher odds of NS. In a structural equation model, we hypothesized that M. perstans infection, higher vitamin E concentration and fewer viral exposures increased the risk of Nodding syndrome while lower vitamin B12 concentration, N. americanus and malaria infections resulted from having Nodding syndrome. We found no evidence that O. volvulus, antileiomodin-1 antibodies, vitamin B6 and other factors were associated with Nodding syndrome. Our results argue against several previous causal hypotheses including O. volvulus. Instead, Nodding syndrome may be caused by a complex interplay between multiple pathogens and nutrient levels. Further studies need to confirm these associations and determine the direction of effect.

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Section: Introductionmentioning

confidence: 99%

Parasitic, bacterial, viral, immune-mediated, metabolic and nutritional factors associated with nodding syndrome

Edridge,

Abd-Elfarag,

Deijs

et al. 2023

Brain Communications

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“…66 Interestingly, a recent association was determined between measles virus seropositivity and the "Nodding syndrome (NS)." 67 NS is likely a form of atonic seizures, clinically manifesting as stereotyped head nodding associated with cognitive decline and is prevalent in Tanzania, Uganda and Sudan. Malnutrition may be the factor leading to reactivation of latent measles virus in the brain.…”

Section: Stereotypies and Repetitive Behaviorsmentioning

confidence: 99%

Movement Disorders in Patients with Subacute Sclerosing Panencephalitis: A Systematic Review

Garg,

Patel,

Sankhla

et al. 2024

Movement Disord Clin Pract

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BackgroundSubacute sclerosing panencephalitis (SSPE) is a complication of measles, occurring after a latency of 4–10 years. It continues to occur in developing countries although resurgence is being reported from developed countries. Characteristic features include progressive neuropsychiatric issues, myoclonus, seizures, movement disorders and visual impairment. Electroencephalography (EEG) typically shows periodic generalized discharges, and elevated CSF anti‐measles antibodies are diagnostic. Movement disorders are being increasingly recognized as part of the clinical spectrum, and range from hyperkinetic (chorea, dystonia, tremor, tics) to hypokinetic (parkinsonism) disorders and ataxia.ObjectivesThis article aims to comprehensively review the spectrum of movement disorders associated with SSPE.MethodsA literature search was conducted in PubMed and EMBASE databases in December 2023 and articles were identified for review.ResultsMovement disorders reported in SSPE included hyperkinetic (chorea, dystonia, tremor and tics), hypokinetic (parkinsonism), ataxia and extraocular movement disorders. Myoclonus, a core clinical feature, was the most frequent “abnormal movement.” Movement disorders were observed in all clinical stages, and could also be a presenting feature, even sans myoclonus. Hyperkinetic movement disorders were more common than hypokinetic movement disorders. An evolution of movement disorders was observed, with ataxia, chorea and dystonia occurring earlier, and parkinsonism later in the disease. Neuroradiological correlates of movement disorders remained unclear.ConclusionA wide spectrum of movement disorders was observed throughout the clinical stages of SSPE. Most data were derived from case reports and small case series. Multicentric longitudinal studies are required to better delineate the spectrum and evolution of movement disorders in SSPE.

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“…This included measles paramyxovirus, which, while previously found to be negatively associated with nodding syndrome in Mundri County, 1 was linked by parental self-report to onset of head nodding among Acholi children, and elevated antibody titres were found in a preliminary study of serum of Ugandan patients with nodding syndrome relative to household and community controls. 13 Measles virus was of potential interest in relation to nodding syndrome because some children with acute infection may, years later, develop subacute sclerosing panencephalitis, a progressive and usually fatal brain disorder also associated with head nodding, neuroinflammation and tau pathology. Rinderpest, the measles-related paramyxovirus, was also of interest in Mundri County because, in 2001, children with nodding syndrome were restricted to ethnic Moru (agropastoralists) and absent from ethnic Dinka people, 1 who herded cattle subject to rinderpest.…”

mentioning

confidence: 99%

New clues to the elusive aetiology of nodding syndrome

Spencer

2023

Brain Communications

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Preliminary seroprevalence study of neurotropic virus antibodies in Nodding syndrome

Cited by 4 publications

References 21 publications

Parasitic, bacterial, viral, immune-mediated, metabolic and nutritional factors associated with nodding syndrome

Parasitic, bacterial, viral, immune-mediated, metabolic and nutritional factors associated with nodding syndrome

Movement Disorders in Patients with Subacute Sclerosing Panencephalitis: A Systematic Review

New clues to the elusive aetiology of nodding syndrome

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