Disability assessment among persons with epilepsy in Mahenge, an onchocerciasis-endemic area in Tanzania: A cross-sectional study.

BACKGROUND: A high prevalence of epilepsy has been observed in the onchocerciasis-endemic focus of Mahenge, Tanzania. This study sought to assess the degree of disability experienced by persons with epilepsy (PWE) in Mahenge and identify associations with sociodemographic and clinical features. METHOD: This cross-sectional study was conducted in Mahenge, Tanzania, between February and July 2020. PWE were recruited from the Mahenge epilepsy clinic and four neighbouring rural villages (Mdindo, Mzogezi, Mzelezi and Sali). Data were collected using the 36-item version of the World Health Organization Disability Assessment Schedule 2.0 (WHODAS 2.0) questionnaire for adults. For children aged 5-17 years, we used the Module on Child Functioning developed by UNICEF and the Washington Group. Questionnaires were administered by trained research assistants. Descriptive statistics were performed, and multivariable analyses (gamma and logistic regressions) were conducted. RESULTS: A total of 321 adults (45.5% males) and 48 children (55.3% males) with epilepsy participated. The overall median WHODAS 2.0 score was 4.8% (IQR: 0.9-18.9). The most affected disability domain was 'participating in the society' (median score: 12.5%, IQR: 0-29.2). Fifteen (31.3%) of the children with epilepsy had a disability in at least one domain of the child functioning module, with the 'accepting change' domain harbouring the highest proportion of disabled children (12.5%). Higher seizure frequency and longer epilepsy duration were associated with more disability. CONCLUSION: PWE in Mahenge experience variable degrees of disability. The affected domains indicate the need for societal rehabilitation of PWE in various community and/or social activities. Peer-support groups were instituted at the study sites to address these needs.

Case definitions for onchocerciasis-associated epilepsy and nodding syndrome: A focused review.

Onchocerciasis-associated epilepsy (OAE) is an important but neglected public health problem in onchocerciasis-endemic areas with insufficient or inadequate onchocerciasis control. Hence, there is a need for an internationally accepted, easy-to-use epidemiological case definition of OAE to identify areas of high Onchocerca volvulus transmission and disease burden requiring treatment and prevention interventions. By including OAE as a manifestation of onchocerciasis, we will considerably improve the accuracy of the overall onchocerciasis disease burden, which is currently underestimated. Hopefully, this will lead to increased interest and funding for onchocerciasis research and control interventions, notably the implementation of more effective elimination measures and treatment and support for affected individuals and their families.

Epilepsy-related stigma and cost in two onchocerciasis-endemic areas in South Sudan: A pilot descriptive study.

INTRODUCTION: Epilepsy is a major public health concern in sub-Saharan Africa, particularly in resource-limited rural villages where persons with epilepsy (PWE) are often confronted with a wide treatment gap, frequent stigma and high cost of epilepsy care. We investigated stigma and economic cost related to epilepsy in the states of Maridi and Amadi in South Sudan, two onchocerciasis endemic areas with high epilepsy prevalence. METHODS: Between November 2019 and February 2020, community-based surveys were conducted in eight villages of Maridi and Amadi States. Consenting PWE were identified via a door-to-door approach, and perceived stigma was assessed using the validated Kilifi stigma scale. Additional data about household income, as well as epilepsy-related direct and indirect costs were collected. RESULTS: 239 PWE were recruited (95 from Maridi, 144 from Amadi). Stigma scores were higher in Maridi compared to Amadi (mean scores: 13.9 vs 6.5, p < 0.001). Mean weighted epilepsy costs per month in Maridi (38.4 USD) were double those observed in Amadi (17.6 USD). The main epilepsy-related expenditure was the purchase of anti-epileptic drugs (AED). Stigma scores correlated with epilepsy cost (Spearman-rho = 0.24, p < 0.001) and were positively associated with traditional medicine use (regression estimate = 1.9; p = 0.027). CONCLUSION: In rural South Sudan, PWE and their families often experienced stigma from the community. Higher perceived stigma was associated with traditional medicine use, which increased the overall cost of epilepsy management. Demystifying epilepsy and making AED more accessible would improve the quality of life of PWE and their families, and reduce the economic burden of epilepsy.

Comprehensive management of epilepsy in onchocerciasis-endemic areas: lessons learnt from community-based surveys.

BACKGROUND: Onchocerciasis-endemic regions are known to have a high epilepsy prevalence. Limited resources in these areas and poor access to healthcare by persons with epilepsy (PWE) result in a wide anti-epileptic treatment gap, poor seizure control and a high burden of seizure-related complications. Recent community-based surveys highlight the need for epilepsy management strategies suitable for remote onchocerciasis-endemic villages to ensure better health outcomes for PWE. In this paper, we propose a feasible approach to manage PWE in such settings. MAIN TEXT: Improved management of PWE in onchocerciasis-endemic areas may be achieved by decentralizing epilepsy care. Simplified approaches for the diagnosis and treatment of epilepsy may be used by non-physicians, under the supervision of physicians or specialists. To reduce the treatment gap, a regular supply of subsidized anti-epileptic drugs (AED) appropriate for different types of onchocerciasis-associated epilepsy should be instituted. Setting up a community-based epilepsy surveillance system will enable early diagnosis and treatment of PWE thereby preventing complications. Community awareness programs on epilepsy must be implemented to reduce stigma and facilitate the social rehabilitation of PWE. Finally, strengthening onchocerciasis elimination programs by optimizing community-directed treatment with ivermectin (CDTI) and considering alternative treatment strategies might reduce the incidence of epilepsy. CONCLUSIONS: A community-based approach with task-shifting of epilepsy care from specialists to non-physician health workers will reduce epilepsy-associated morbidity. Increased advocacy and collaboration with various stakeholders is needed to establish a sustainable, cost-effective chronic care model for epilepsy that will significantly improve the quality of life of PWE in onchocerciasis-endemic regions.

Community perceptions of epilepsy and its treatment in an onchocerciasis endemic region in Ituri, Democratic Republic of Congo.

BACKGROUND: A recent study in the Logo and Rethy health zones in the Ituri Province in the Democratic Republic of Congo (DRC) reported that the majority of the persons with epilepsy (PWE) had not been treated with anti-epileptic medication (AEM) or had stopped treatment. Prior to the implementation of an epilepsy treatment programme in these health zones, this study investigated the perceptions and experiences regarding epilepsy and its treatment amongst community leaders, PWE and/or their families, traditional healers and health professionals. METHODS: A total of 14 focus group discussions (FGD) and 39 semi-structured interviews (SSI) were conducted with PWE and/or their family members, community leaders, traditional healers, and health professionals in the Logo and Rethy health zones during February 2-19, 2017. RESULTS: In the two health zones, the clinical signs of convulsive epilepsy were recognized by community members. However, a variety of misconceptions about epilepsy were identified including the beliefs that epilepsy is a family-related condition, a contagious disease, is transmitted by insects, saliva or by touching a person of the same sex during seizures, or is caused by evil spirits and witchcraft. The role of traditional healers in spreading these beliefs was revealed. The study also reported sexual abuse on PWE, stigmatisation of PWE and loss of productivity of PWE and their families. Some PWE had been using AEM and traditional treatment but were not convinced about the efficacy of these treatment options. The lack of training of health providers about epilepsy care, financial barriers in obtaining AEM, and the shortage of AEM at primary health facilities were revealed. As remedies, the community requested access to a decentralized center for epilepsy treatment. They also proposed using churches and community health workers as communication channels for information about epilepsy. CONCLUSIONS: Clinical signs of convulsive epilepsy were recognized by the community in the Logo and Rethy health zones but many misconceptions about epilepsy were identified. A comprehensive community-based epilepsy treatment programme with an affordable uninterrupted AEM supply needs to be established. Such a programme should address stigma, misconceptions, sexual abuse and foster the rehabilitation of PWE to alleviate poverty.

Burden of onchocerciasis-associated epilepsy: first estimates and research priorities.

BACKGROUND: Since the 1990s, evidence has accumulated of an increased prevalence of epilepsy in onchocerciasis-endemic areas in Africa as compared to onchocerciasis-free areas. Although the causal relationship between onchocerciasis and epilepsy has yet to be proven, there is likely an association. Here we discuss the need for disease burden estimates of onchocerciasis-associated epilepsy (OAE), provide them, detail how such estimates should be refined, and discuss the socioeconomic impact of OAE, including a cost-estimate for anti-epileptic drugs. MAIN BODY: Providing OAE burden estimates may aid prevention of epilepsy in onchocerciasis- endemic areas by inciting and informing collaboration between onchocerciasis control programmes and mental health services. Epilepsy not only massively impacts the health of those affected, but it also carries a high socioeconomic burden for the households and communities involved. We used previously published geospatial estimates of onchocerciasis in Africa and a separately published logistic regression model quantifying the association between onchocerciasis and epilepsy to estimate the number of OAE cases. We then applied disability weights for epilepsy to quantify the burden in terms of years of life lived with disability (YLD) and estimate the cost of treatment. We estimate that in 2015 roughly 117 000 people were affected by OAE across onchocerciasis-endemic areas previously under the African Programme for Onchocerciases control (APOC) mandate where OAE has ever been reported or suspected, and another 264 000 persons in onchocerciasis-endemic areas where OAE has never been investigated before. The total number of YLDs due to OAE was 39 300 and 88 700 in these areas respectively, based on a weighted mean disability weight of 0.336. The burden of OAE is approximately 13% of the total YLDs attributable to onchocerciasis and 10% of total YLDs attributable to epilepsy. We estimated that by 2015 the total costs of treatment with anti-epileptic drug for OAE cases would have been a minimum of 12.4 million US$. CONCLUSIONS: These estimates suggest a considerable health, social and economic burden of OAE in Africa. The treatment and care for people with epilepsy, especially in hyperendemic onchocerciasis areas with high epilepsy prevalence thus requires more financial and human resources.

Diagnosing risk factors alongside mass drug administration using serial diagnostic tests-which test first?

Background: When tests are used in series to determine individual risk factors and infection status in a mass drug administration (MDA), the diagnostics, test order and subsequent treatment decisions (the testing algorithm) affect population-level treatment coverage and cost, but there is no existing framework for evaluating which algorithm optimizes any given outcome. Methods: We present a mathematical tool (with spreadsheet implementation) to analyse the effect of test ordering, illustrated using treatment for onchocerciasis in an area where high-burden Loa loa co-infections present a known risk factor. Results: The prevalence of the infection and risk factor have a non-linear impact on the optimal ordering of tests. Testing for the MDA infection first always leaves more infected people untreated but fewer people with the risk factor being misclassified. The cost of the treatment given to infected individuals with the risk factor does not affect which algorithm is more cost effective. Conclusions: For a given test and treat algorithm and its costs, the correct strategy depends on the expected prevalence. In most cases, when the apparent prevalence of the target infection is greater than the apparent prevalence of the risk factor, it is cheaper to do the risk factor test first, and vice versa.

Report of the first international workshop on onchocerciasis-associated epilepsy.

BACKGROUND: Recently, several epidemiological studies performed in Onchocerca volvulus-endemic regions have suggested that onchocerciasis-associated epilepsy (OAE) may constitute an important but neglected public health problem in many countries where onchocerciasis is still endemic. MAIN TEXT: On October 12-14th 2017, the first international workshop on onchocerciasis-associated epilepsy (OAE) was held in Antwerp, Belgium. The workshop was attended by 79 participants from 20 different countries. Recent research findings strongly suggest that O. volvulus is an important contributor to epilepsy, particularly in meso- and hyperendemic areas for onchocerciasis. Infection with O. volvulus is associated with a spectrum of epileptic seizures, mainly generalised tonic-clonic seizures but also atonic neck seizures (nodding), and stunted growth. OAE is characterised by an onset of seizures between the ages of 3-18 years. Multidisciplinary working groups discussed topics such as how to 1) strengthen the evidence for an association between onchocerciasis and epilepsy, 2) determine the burden of disease caused by OAE, 3) prevent OAE, 4) improve the treatment/care for persons with OAE and affected families, 5) identify the pathophysiological mechanism of OAE, and 6) deal with misconceptions, stigma, discrimination and gender violence associated with OAE. An OAE Alliance was created to increase awareness about OAE and its public health importance, stimulate research and disseminate research findings, and create partnerships between OAE researchers, communities, advocacy groups, ministries of health, non-governmental organisations, the pharmaceutical industry and funding organizations. CONCLUSIONS: Although the exact pathophysiological mechanism underlying OAE remains unknown, there is increasing evidence that by controlling and eliminating onchocerciasis, OAE will also disappear. Therefore, OAE constitutes an additional argument for strengthening onchocerciasis elimination efforts. Given the high numbers of people with epilepsy in O. volvulus-endemic regions, more advocacy is urgently needed to provide anti-epileptic treatment to improve the quality of life of these individuals and their families.

Rapid assessment method for prevalence and intensity of Loa loa infection.

OBJECTIVE: To assess the validity of observations on eye worm and Calabar swellings for the rapid assessment of the prevalence and intensity of loiasis at the community level. METHOD: A total of 12895 individuals over the age of 15 years living in 102 communities in Cameroon and Nigeria took part in the study. A standardized questionnaire was administered to participants from whom finger-prick blood samples were collected and examined for Loa loa microfilariae. Rapid assessments of the prevalence and intensity of loiasis were made on the basis of a history of eye worm or Calabar swellings. FINDINGS: There was a strong correlation between the indices of the rapid assessment procedures and the parasitological indices of L. loa endemicity. The rapid assessment indices were effective in diagnosing high-risk communities (sensitivity 94-100%; specificity 66-92%). The highest sensitivity (100%) and specificity (92%) were obtained with a rapid assessment procedure based on a history of eye worm lasting 1-7 days together with confirmation by the guided recognition of a photograph of adult L. loa in the eye. CONCLUSION: Rapid assessment of the prevalence and intensity of loiasis at the community level can be achieved using a procedure based on the history of eye worm lasting 1-7 days together with confirmation by the guided recognition of a photograph of an adult L. loa in the eye.

Independent evaluation of onchocerciasis rapid assessment methods in Benue State, Nigeria.

OBJECTIVE: To evaluate the prevalence of palpable nodules or skin depigmentation as rapid indicators of onchocerciasis epidemicity in at-risk communities. METHOD: We examined data collected in Benue State on 11035 individuals in 32 villages to evaluate these rapid assessment methods. RESULTS: The prevalence of palpable nodules correlates more closely with microfilarial prevalence (r=0.68, P<0.001) and community microfilarial load (r=0.64, P<0.001) than the prevalences of skin depigmentation or other potential rapid indicators. The recommended cut-off value for palpable nodules of 20% or more in males aged >20 years had a sensitivity of 94% and specificity of 50% compared to a cut-off of 40% or more for microfilarial prevalence in all ages. This would mean that in these 32 villages 17 of 18 would have been correctly identified for treatment, and a further 7 at lesser risk would have been targeted for treatment. CONCLUSIONS: Skin snipping and parasitological examination can be replaced by the simpler method of palpating onchocercal nodules to identify communities at serious risk of onchocerciasis. This has important operational benefits for onchocerciasis control programmes.

Ivermectin distribution and the cultural context of forest onchocerciasis in South Province, Cameroon.

This investigation examined the cultural context of forest onchocerciasis in several communities in the Dja-Lobo Division of southern Cameroon. The study sought to elucidate behaviors that would enhance or diminish health status relative to forest onchocerciasis and other filarial infections, and to make culturally sensitive and appropriate recommendations regarding the development of health education materials and the long-term sustainability of the ivermectin distribution program in Dja-Lobo. The study consisted of two sequential components; the first was a qualitative study of a few severely affected villages and the second was a quantitative study of 212 randomly selected heads of households from eight villages. The Boulou and Baka peoples in these communities defined general filariasis (minak) as small worms under the skin, identified flies as important transmitters of the illness, and indicated that blindness and other skin and ocular problems were a consequence of the illness. Illness of the Dja (referring to an illness found near the Dja River) was another illness that was closely linked to onchocerciasis; local people indicated it was transmitted by the black flies found near the Dja River, resulting in severe itching and leopard skin. These and other cultural-behavioral data on filariasis were used to implement a health education and distribution program.

PIP: The cultural context of forest onchocerciasis was studied in the Boulou and Baka ethnic communities in the Dja-Lobo Division of southern Cameroon. A 2-day survey used focus group interviews followed by a questionnaire administered to 212 randomly selected individuals in 8 communities (88 male and 124 females heads of household) to assess their knowledge about onchocerciasis. Most people (98%) had some knowledge about the disease. Minak was the term used for filariasis by most people (97%) and people knew (90%) that black fly (nyamendimi) was responsible for its transmission. Other vectors of the illness identified were mosquitoes, dirty water, sorcery, and taboo foods. 81% thought that maternal transmission was possible and 66% indicated that filariasis could be transmitted sexually. Virtually all respondents associated itching and rash with minak (filariasis) and more than 60% also recognized the swelling of the skin and leopard skin as manifestations of filariasis. Filariasis, malaria, worms, and blindness were placed in the middle category when the severity of various diseases was ranked by 20 Boulou adults. In contrast, the Baka did not think that filariasis caused blindness, nor that it is linked to eye-worms. However, the 212 individuals ranked blindness as the most severe among other diseases (filaria, malaria, diarrhea, and intestinal worms). 80% of the Boulou and Baka adults had had filariasis in the previous year, but only 5% of the Boulou children and none of the Baka children had had filariasis during that time period. With respect to intestinal worms, 71% of the Boulou adults and 60% of the Baka adults had had intestinal worms in the previous year, while more than 90% of the Boulou children and all of the Baka children had had intestinal worms. Of the 90% who revealed that they had had filariasis at least once before, 69% sought treatment. 54% had tried traditional treatment, while 50% had tried Notezine, 49% had tried Phenergan, and 38% had tried M.G. Lumiere.

Prevalence of depigmentation of the shins: a simple and cheap way to screen for severe endemic onchocerciasis in Africa.

The prevalence of skin depigmentation in the pretibial region is closely correlated in the adult Congolese population with conventional indices (microfilarial index, mean microfilarial density, cyst index) for the evaluation of endemic onchocerciasis. Individuals over 15 years of age in endemic villages who had a microfilarial index of 80-90%, had a cystic index of 60-70% and a pretibial depigmentation index about 20% (30-40% for individuals aged > 50 years). Thus, in Africa, this new evaluation index offers a simple, noninvasive, quick and cheap way to identify areas where the level of endemic onchocerciasis is probably high and which are likely to benefit from an anti-onchocerciasis campaign.

PIP: Skin depigmentation is a classic diagnostic sign of onchocerciasis and presents typically as an irregular, flat spot surrounded by slightly hyperpigmented zones. The depigmentation occurs mainly on the shins. The prevalence of skin depigmentation has been proposed as a marker for evaluating the endemicity of onchocerciasis in Nigeria, but has been contested for use in South America on the grounds of non-specificity. The microfilarial index currently used to assess the level of onchocerciasis endemicity is relatively time-consuming, costly, and harbors the risk of transmitting viral infections during mass screenings. 991 people over 15 years of age of mean age 55.4 years in five villages in the Congo endemic for onchocerciasis participated in this study. Individuals who had a microfilarial index of 80-90% had a cystic index of 60-70% and a pretibial depigmentation index of approximately 20%. The authors conclude that the existence of shin depigmentation as an evaluation index in Africa offers a simple, noninvasive, quick, and inexpensive way to identify areas where the level of endemic onchocerciasis is probably high and which are likely to benefit from an anti-onchocerciasis campaign. Although the new index does not allow total specificity, the incidence of shin depigmentation among patients with African onchocerciasis is such that there is probably little risk of confusing holo- or hyperendemic zones with an area where leprosy or treponema are endemic. In the absence of onchocerciasis, bites from Simulium spp. flies may also be responsible for skin depigmentation, as may be streptocerciasis. In this latter condition, however, the predominant site of depigmentation is not the shin. The authors have observed no notable prevalence of shin depigmentation in northern Congo, which is infested with S. albivirgulatum flies, but with no onchocerciasis transmission, and where there are also zones of streptocerciasis.

Musculo-skeletal pain (MSP) in onchocerciasis: a potential in the rapid low cost epidemiological survey and in the assessment of impact of ivermectin treatment on a community.

The diagnostic potential of musculo-skeletal pain (MSP) in the rapid assessment of onchocerciasis of morbidity and of the impact of ivermectin treatment in a community is brought to picture. The clinical impression is that MSP is a common, early, chronic important symptom of onchocerciasis. With a significant association with onchocerciasis, MSP has the potential of being an important operational diagnostic clue in the detection of onchocerciasis. It has the advantage of being easily recognizable, and therefore can be applied by primary health care workers. MPS is proved to cause the greatest morbidity, particularly among working farmers in areas where the forest strain of O. volvulus is predominant. Thus, onchocerciasis is contributing more than was perhaps realized to the reduced output of work from farmers. However, MPS, a parasitic rheumatism, is proved to be eminently susceptible to treatment by DEC.

The economics of blindness prevention in Upper Volta under the Onchocerciasis Control Program.

This paper proposes a new approach to the economics of blindness prevention under the Onchocerciasis Control Program in Upper Volta. It differs from previous economic analyses of onchocerciasis control in three important respects. First, it uses empirical data as the basis of an estimate of the epidemiological effectiveness of the intervention. Second, it focuses on the prevention of permanent disability and premature death due to onchocercal blindness as the major health improvement attributable to onchocerciasis control. Third, it emphasizes cost-effectiveness rather than cost-benefit analysis. This limitation is imposed by the difficulty of undertaking a comprehensive assessment of the benefits of onchocerciasis control. In particular, the extent to which control of partial visual impairment and infection without ocular involvement would increase the effective supply of labor, and also the extent to which control would increase the effective supply of land by inducing new settlement in the river valleys, have not been clearly established. The cost-effectiveness approach is limited because it foregoes the opportunity provided by cost-benefit analysis to compare the relative desirability of investing in onchocerciasis control with alternative investments in other sectors. However, it does permit useful judgments to be made about the relative efficiency of allocating scarce resources to onchocerciasis control compared to other possible investments within the health sector. For this purpose, an illustrative comparison is made with estimates of the cost-effectiveness of measles immunization.

Cost-effectiveness of blindness prevention by the Onchocerciasis Control Programme in Upper Volta.

The article presents a cost-effectiveness analysis of the Onchocerciasis Control Programme in Upper Volta. The analysis uses a new approach to the measurement of health project effectiveness, by considering the number of healthy years of life added by the prevention of permanent disability and premature death attributable to onchocercal blindness. The approach emphasizes the central role of social value judgements in allocating health resources-in particular the relative weights assigned to preventing disability and postponing death, present and future health benefits, and health gains among productive and non-productive individuals. The quantitative results yield the following cost-effectiveness estimates for blindness prevention through onchocerciasis control: US$20 per year of healthy life and per productive year of healthy life added, and US$150 per discounted year of healthy life and per discounted productive year of healthy life added. As an illustrative example, a comparison is made with estimates of the cost-effectiveness of measles immunization.

Babana Parasitic Diseases Project. I. The study area and a preliminary assessment of onchocercal endemicity based on the prevalence of "leopard skin".

A prevalence survey of "leopard skin" (LS) was carried out in 24 villages to assess the distribution of onchocerciasis in the Babana District of Nigeria. The findings suggested that onchocerciasis is highly endemic in this community. Of 1,310 adults examined for the presence of the characteristic onchocercal depigmentation, 26.4% were found affected. LS occurred more frequently among the cattle Fulani people than among other local ethnic groups (P less than 0.001) and was significantly more commonly associated with the left than with the right leg (P less than 0.001). A background of the study area is given.