Amaral Luís-Jorge, Jada Stephen Raimon, Carter Jane Y, Bol Yak Yak, Basáñez María-Gloria, Newton Charles R, Fodjo Joseph N Siewe, Colebunders Robert
Global Health Institute, University of Antwerp, Antwerp, Belgium.
UK Medical Research Council Centre for Global Infectious Disease Analysis, and London Centre for Neglected Tropical Disease Research, Department of Infectious Disease Epidemiology, School of Public Health, Imperial College London, London, United Kingdom.
PLoS Negl Trop Dis. 2025 Jun 30;19(6):e0013244. doi: 10.1371/journal.pntd.0013244. eCollection 2025 Jun.
Epilepsy is a major health concern in onchocerciasis-endemic regions with intense transmission, where the infection is associated with a high epilepsy burden. This study investigated epilepsy prevalence and mortality in five onchocerciasis-endemic counties of South Sudan, and the association between onchocerciasis transmission and epilepsy, including probable nodding syndrome (pNS).
House-to-house cross-sectional surveys (2021-2024) identified persons with suspected epilepsy (sPWE) and retrospectively documented deaths among sPWE and individuals without epilepsy (IWE). Epilepsy diagnoses, including pNS, were confirmed by trained clinicians. Ongoing transmission was assessed using anti-Ov16 seroprevalence in children aged 3‒9 years. Age- and sex-standardised epilepsy, pNS and anti-Ov16 prevalence were calculated, along with age- and sex-standardised mortality rates and standardised mortality ratios (SMRs) with 95% confidence intervals (95%CIs), using IWE as the reference population. Weighted arcsin-transformed linear regression was used to explore the association between epilepsy and anti-Ov16 prevalence.
Among 34,019 individuals screened, 166 deaths occurred in 3,101 person-years for sPWE versus 466 deaths in 63,420 person-years for IWE. Epilepsy prevalence was 4.1% (range: 2.3-7.1%), and pNS prevalence was 1.5% (range: 0.6-2.2%). Anti-Ov16 seroprevalence among children was 23.3% (range: 1.4-44.1%). Each 1.0 percentage point increase in standardised anti-Ov16 seroprevalence was statistically significantly associated with an average rise of 0.10 percentage points in standardised epilepsy prevalence and 0.04 percentage points in standardised pNS prevalence. Median age at death was lower for sPWE (20 years) than IWE (38 years; Mann-Whitney U-test p-value < 0.0001). Standardised mortality rates per 1,000 person-years were statistically significantly higher in sPWE (67.6, 95%CI: 52.6-87.1) than in IWE (9.0, 95%CI: 7.8-10.3). The overall SMR was 6.9 (95%CI: 5.9-8.0), indicating sPWE were seven times more likely to die than IWE.
The high epilepsy burden in onchocerciasis-endemic areas is driven by elevated epilepsy prevalence and mortality. Integrated onchocerciasis and epilepsy programmes must be strengthened to decrease epilepsy incidence and ensure uninterrupted access to antiseizure medication.
在盘尾丝虫病高传播流行地区，癫痫是一个主要的健康问题，在这些地区，感染与高癫痫负担相关。本研究调查了南苏丹五个盘尾丝虫病流行县的癫痫患病率和死亡率，以及盘尾丝虫病传播与癫痫（包括可能的点头综合征[pNS]）之间的关联。
逐户横断面调查（2021 - 2024年）确定疑似癫痫患者（sPWE），并回顾性记录sPWE和无癫痫个体（IWE）中的死亡情况。包括pNS在内的癫痫诊断由经过培训的临床医生确认。通过检测3至9岁儿童的抗Ov16血清阳性率来评估持续传播情况。计算年龄和性别标准化的癫痫、pNS和抗Ov16患病率，以及年龄和性别标准化死亡率和标准化死亡比（SMR）及其95%置信区间（95%CI），以IWE作为参考人群。使用加权反正弦变换线性回归来探讨癫痫与抗Ov16患病率之间的关联。
在34,019名筛查个体中，sPWE在3101人年中有166例死亡，而IWE在63,420人年中有466例死亡。癫痫患病率为4.1%（范围：2.3 - 7.1%），pNS患病率为1.5%（范围：0.6 - 2.2%）。儿童抗Ov16血清阳性率为23.3%（范围：1.4 - 44.1%）。标准化抗Ov16血清阳性率每增加1.0个百分点，标准化癫痫患病率平均上升0.10个百分点，标准化pNS患病率平均上升0.04个百分点，差异有统计学意义。sPWE的中位死亡年龄（20岁）低于IWE（38岁；Mann - Whitney U检验p值<0.0001）。每1000人年的标准化死亡率，sPWE（67.6，95%CI：52.6 - 87.1）显著高于IWE（9.0，95%CI：7.8 - 10.3）。总体SMR为6.9（95%CI：5.9 - 8.0），表明sPWE死亡的可能性是IWE的7倍。
盘尾丝虫病流行地区的高癫痫负担是由癫痫患病率和死亡率升高所致。必须加强盘尾丝虫病和癫痫综合防治项目，以降低癫痫发病率，并确保不间断地获得抗癫痫药物。
