Study site and population

Ethnographic research was carried out in Northern Uganda between 2015 and 2017, primarily in Kitgum district and partly in Gulu district. These districts were selected purposively on the basis of their having NS affected and non-affected villages. In addition, the research team was introduced to these districts during the second NS conference held in Gulu and thus leveraged on linkages with district officials and Hope4Humans to focus the study in the two districts. Extending the study to other districts like Pader and Lamwo with similar ethnic composition as the study areas was not feasible. However, focusing on the two districts enabled us to visit the research subjects repeatedly over the three-year period (2015–2017) which facilitated gaining community trust and an in-depth understanding of the context and beliefs around NS.

The recent history of Northern Uganda, and our field sites, was dominated by the war between the Lord’s Resistance Army (LRA) and the government of Uganda, which lasted from 1987 to 2006. The forced displacement of much of the population into IDP camps took place from 1996–2007 where people were subjected to violence and governmental surveillance [29, 30]. Prior to the LRA war, Northern Uganda had endured over three decades of civil conflict, which enhanced the already existing North-South divide between the Acholi and Langi ethnic groups of the North and the Bantu in the South [31].

Political distrust and suspicion about perceived neglect by the current government towards the Acholi people is a recurrent sentiment that has prevailed through all three epidemics–Ebola, HIV [32] and now NS. President Museveni came to power by overthrowing an Acholi leader, General Tito Okello Lutwa in 1986. As a counterinsurgency, the LRA war against president Museveni and his National Resistance Army [33] began a year later, mainly in response to alleged atrocities by the NRA against the Acholi who had feared retribution by the new government [32]. These feelings have been reinforced by the level of economic investment, governmental and non-governmental development initiatives, social services and business-opportunities available in the south of the country as compared to the substantially less developed northern region [32]. When responding to disease outbreaks in Northern Uganda it is crucial to keep this historical political context in mind.

In 2015, a study retrospectively reported that the first case of NS was seen in Western Uganda in 1994 [34], however, it was only after media uproar in 2012 that NS attracted national attention through a campaign of the opposition party [3] highlighting the perceived neglect of healthcare in the North [3, 35]. Consequent parliamentary debates about the outbreak of a previously unknown ‘disease’ named “Nodding Syndrome” drove governmental and non-governmental organisations (NGOs) to provide services and resources for NS control [3]. These consisted of free of charge provision of anti-epileptic drugs (AED) for the management of seizures, along with nutritional supplements as well as behavioural and physical therapy at 17 government health facilities in the affected region [19]. However, the resources provision for the NS response have been inadequate for different reasons.

Between 2012 and 2017, NS outreach services and comprehensive in-patient care for severely affected children were also provided by a US funded NGO, Hope for Humans in Gulu district. In late 2017 the NGO had to close down after several failed efforts of finding a sustainable stream of funding or governmental support. This closure led to renewed accusations and media reports of the government’s and ministry of health’s failure to prioritize and protect Acholi children. After much political debate the new government committed to improved NS service provision in Northern Uganda. There has been a cyclical trend of political propaganda, where the Acholi in the north, both politicians and civilians, have criticized the ruling government of inadequate support, which has driven governmental interventions for NS in the region.

The NS epidemic affected an already impoverished region of the country, exacerbating the economic burden on caretakers and the community [28, 36]. The region is still one of the most impoverished regions of the country. While 85% of Uganda’s poor were reported to live in the Northern and Eastern regions of the country in 2013, the Northern region accounted for more than half [37]. The villages we visited in Gulu and Kitgum districts, survived mainly on subsistence farming. Droughts and el niño rains periodically affected the food production and inhabitants faced severe food shortages.

Village inhabitants were mostly of the Acholi ethnic group, who had lived in the area for several generations but were displaced from their homes during the LRA war, when they lived in IDP camps [29].

Sampling

Contrasting villages with high prevalence (more than 6%) and low prevalence (about 1%) of epilepsy/Nodding syndrome were purposefully selected. Prevalence was calculated based on the 2012 data provided by the ministry of health for all villages in the Kitgum district, and from the Hope4Humans estimates and the local health centre for Gulu district. The study began with an exploratory phase, where data was collected from two high prevalence (Akoyo and Ajan) and one low prevalence (Dino) village in Gulu district, and from three villages in Kitgum district. Health officials at the district hospital which had a ward that handled epilepsy and nodding syndrome cases were interviewed. The main phase of data collection focused on Kitgum district as we were able to find 10 contrasting villages within two sub-counties, Akwang and Amida, which made it relevant for our study. Villages selected in Kitgum district are listed in Table 1. All villages were classified as hyper-endemic for onchocerciasis.

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Table 1. Prevalence of Nodding Syndrome & epilepsy in selected villages in Kitgum district.

https://doi.org/10.1371/journal.pntd.0007344.t001

Participant recruitment was carried out through a mixed-sampling approach, including both theoretical (based on emergent findings) and snowball sampling techniques (relying on one sampled person to contact the next person in the sample) in order to have maximum variation and representativeness in the sample. We purposefully selected varying social groups based on age; gender; persons directly affected/unaffected by NS/Epilepsy; occupation and livelihood regardless of local hierarchy or locally perceived expertise as shown in Table 2.

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Table 2. Respondents.

https://doi.org/10.1371/journal.pntd.0007344.t002

Data collection

An ethnographic approach, triangulating participant observations, in-depth interviews, focus group discussions, was chosen because it allows in-depth descriptions of disease perceptions and the detection of unforeseen and unknown variables that are hard to measure quantitatively [38]. Additionally, text analysis of media reports was used to deepen the contextualization of the political environment and cross-check emerging themes. Data collection centred around emergent themes such as perceptions of epilepsy and the experiences of those affected, access to and perceptions of public, private and traditional health facilities in general and specifically for NS/epilepsy, health seeking behaviour, religious practices, daily activities and livelihoods.

Participant observation was used to acquire an understanding of the local context. The researchers participated in daily activities in the community and the home setting, observed and participated in events in their usual context, while having informal conversations with a variety of community members. This method was used to cross-check the validity of information obtained in semi-structured interviews by establishing rapport and building trust with community members to reduce response bias. Repeated visits were made to the selected villages over the course of two years. Brief notes were taken during informal interviews if appropriate, otherwise the conversations were written down in detail afterwards.

94 in-depth interviews were conducted at participants’ homes in private or in places where the respondent felt at ease. Most in-depth interviews took an hour and were audio recorded with consent and conducted in the local language, Acholi. If the respondent did not wish to be audio recorded, detailed hand-written notes were taken. Later, all recordings were transcribed verbatim in Acholi and translated in English by trained field assistants.

13 formal focus group discussions were conducted (12 in Kitgum and 1 in Gulu) mostly at village centres, at market places or outside people’s homes when people were gathered together to socialise or for an event or they were organised by the village leaders in advance. Group discussions were used for different purposes: i.e., to get a quick picture of the village activities, livelihoods and facilities available like health centres, schools, traditional healers, water access points, and with village elders to get a better idea of how things might or might not have changed in contrast to the past, with the same gender to understand social norms, gender roles and disease perceptions. Some group discussions were audio recorded with consent while some were documented with hand written notes. All discussions were conducted in the local language, Acholi. Like IDIs, all recordings were transcribed verbatim in Acholi and translated to English by trained field assistants.

Articles in media were monitored from 2015–2017 by using ‘google alerts’ service, i.e. a notification tool offered by Google based on set keywords, for the keywords ‘nodding disease’ and ‘nodding syndrome’ respectively. Most articles came from online English newspapers of Uganda, like “The observer”, “The monitor”, “Daily monitor” and “Acholi times” and occasionally there were some from “Huffington post” and scientific journals. Earlier articles dating back to 2012 were searched for through a google search. All articles were used to assess what was being presented in the media and analysed comparatively with emerging themes from the field.

Data analysis

Data analysis was retroductive (combining inductive analysis of field data and theory from existing anthropological literature)[39] and was carried out concurrently with data collection. Relevant categories for analysis were identified combining data from participant observation, interviews and group discussions. Through the analysis process, data were constantly subjected to theoretical perspectives so as to ensure theoretical triangulation and to embed the findings in existing literature. We used concepts of uncertainty and meaning making coupled with sense making of epilepsy as elaborated by Susan Whyte [40, 41]. We also looked at fear and distrust and its role in the creation of rumours [42], also in conjunction with other epidemics that occurred in the same area to conceptualise the unique socio-political context [32, 43, 44], annihilation anxieties of minority and marginalised populations [45]. Data were imported, managed and analysed using NVivo 12 Qualitative Data Analysis software (QSR International Pty Ltd. Cardigan UK).

Ethical considerations

The study was reviewed and approved by the Institutional Review Board of the Institute of Tropical Medicine in Antwerp (IRB/AB/ac/036 Ref: 983/14); the Makerere University School of Medicine’s Research Ethics Committee (REC REF 2015–079) and the Uganda National Council for Science and Technology (REF: SS 3845). Oral consent was obtained from all participants and preferred, since the act of signing one’s name when providing information can be a reason for mistrust, particularly among illiterate populations, which can significantly reduce the quality of the data (Refer to justification of oral consent attached). Oral consent was documented by the interviewer on a separate paper form. Interviewers followed the European Guidelines for FP7 projects and the guidelines of the American Anthropological Association.

Children who are able to give assent to participate in research were asked to provide assent in addition to the consent of the legally authorized representative. Young people aged 16–18 with sufficient understanding were asked their full consent to participate in research independently of their parents and guardians. If a potential respondent was cognitively impaired, a close family member was asked for consent to ask questions pertinent to the potential respondent. Confidentiality was maintained by storing all data on a password protected computer and not transcribing names of respondents. Findings from the study will be disseminated in the form of scientific manuscripts which will be shared with the Makerere University School of Medicine’s Research Ethics Committee and the Uganda National Council for Science and Technology and the Ministry of Health.

Making sense of a ‘new’ disease in a politically contested, war-torn area

Given the timing of the NS epidemic, community members linked the aetiology of NS to the LRA war in several ways. The most commonly perceived cause was poisoning either through the pollution of air and water with gunpowder during the war or by toxins in the food distributed at IDP camps. The second most commonly stated cause was that the spirits of the deceased during the war were angry for not getting a respectful burial and so were taking revenge on their children by inflicting this disease on them. Illness interpretations also included perceptions related to the epidemic occurrence of the disease outbreak. Crowding was seen to have enhanced the spread of the disease, ‘like measles, through the air’ due to the high concentration of people in the camps. The perceived epidemic nature of NS also led to some not believing that it was a form of epilepsy, an illness well known to them as illustrated in the following quote: “You researchers say this is epilepsy, but we know epilepsy … we used to have it before but it was only one or two persons in the entire village who were affected… but this is different. This came during the war, and this affects entire villages. This is not epilepsy, this is because of the war. (…) Why is it that the villages where most atrocities took place, are also the villages that have the most people affected (by NS)?” (IDI, father of NS victim).

Some associated NS with changes in political power, as illustrated at the funeral of an NS victim: “During Obote’s first regime, there was a disease called ‘two nailon’ [gonorrhoea]. Later he was over thrown by Amin and this sickness disappeared … When Obote came back to power again, there came HIV/AIDS and this continued in Museveni’s regime. And in Museveni’s government there comes this sickness, this strange disease called nodding syndrome.” (Sermon of a middle-aged Acholi Catholic priest)

Many respondents had also heard about the blackfly as a potential cause from researchers and health officials. However, few agreed with this possibility. Many were not convinced as they logically reasoned that blackflies had always been around while NS was something new that had started occurring during the war.

Some resented and distrusted the government and suspected they were the reason for this illness. This distrust was due to the perceived lack of response to the NS outbreak until 2012—almost 15 years after the first cases were noticed, and approximately six years after the war had ended. The government response was perceived as having been initiated after the political propaganda around the disease began circulating through the media attention.

Besides the growing sense of suspicion towards the government, Box 1 conveys the sentiment that governmental authorities did not want affected locals to say that NS existed and was increasing. This resentment explains the second uproar in the media in 2018, after the closing of the NGO, Hope for Humans. Hope for Humans was requesting governmental funds and encouraging the government to take over the responsibility. When this request was refused the NGO was unsustainable and closed down. It was evident that people felt that the government and the MoH were not acknowledging the severity of the problem. The media reported that there was an angry backlash from the northern political representatives when the MoH suggested that there were no more new cases of NS while their people strongly asserted that there were in fact new cases. While the MoH may not be wrong to say that there were no new cases of NS according to case definitions, community members still perceived all epilepsies that occurred during or after the war to be NS and thus thought that the MoH was covering up facts and dismissing the problems people in the north were facing.

The political propaganda that drove the initial response to the outbreak influenced how NS became a tool for power both for politicians and for people in the community gaining resources through the epidemic. Box 2 shows that when these services were abruptly stopped, feelings of resentment and distrust towards the government were reinforced.

However, some still had a different view of the government. While most community members were disheartened by the prospects of NS and felt angered, frustrated and neglected by the government, some believed that researchers coming to the area were government agents who were not trying to find a remedy for NS.

Consequences for health interventions

The perceived link between NS, the war, forced displacement to government-protected camps, in addition to the overwhelming burden of caretaking under extreme poverty, the social inequality and ethnic tensions all enhanced distrust of the government. These factors directly created barriers to NS service delivery in terms of limiting care available in the private sector, increased distrust and reluctance to accept treatment and prevention for onchocerciasis, all relevant for managing NS.

Some non-governmental health facilities, for example, the local Catholic missionary St. Joseph’s hospital in Kitgum, prefered not to treat NS patients due to the political nature of the disease. Reportedly, during the initial period when NS was getting political and media attention, the missionary hospital hosted a researcher who independently observed NS without informing them. After visiting one of the affected villages by himself, news reached the authorities and police tracked him down for questioning. The foreigner was reportedly arrested and deported for not following proper protocol and hospital employees were questioned. Whatever the veracity of the details of this and similar stories, it has led to sensitivity around accepting NS patients as illustrated by the following quote: “We care for epileptic patients in this facility but for NS patients, we always refer them to Kitgum Government Hospital because NS has ‘political affiliations’, and we are faith-based so we don’t want to mix politics and religion.” (IDI, hospital staff) What is interesting however, is how NS is distinguished from other epilepsies at hospitals, where NS and epilepsy were considered two separate medical conditions, whereas people in the community, perceived all epilepsies that occurred during or after the war as NS.

Additionally, news of incidents like this spread through the community quickly and like any story travelling through the grapevine, it lost or enhanced some details allowing differing interpretations to arise. Some became suspicious of secrecy they observed or by the hidden agendas of researchers, the government or both.

Despite the distrust of the government, most NS affected families still sought care in government facilities. While this behavior was seemingly counterintuitive, there were two main reasons for people to do so. Firstly, people were desperate for a cure as the burden of this disease was immense. Besides biomedical treatments, desperation had driven people to seek additional treatment options, including an array of traditional remedies through herbalists, spiritual healers and religious healers. This desperation was illustrated by a father who lost his son to NS. He expressed emphatically, “Those who died are the ones that are cured” implying that death among children with NS was often perceived as better than the continued suffering and disease-related hopelessness associated with NS within the context of poverty. Anti-epileptic drugs, when available, helped suppress symptoms for most patients and when the perceived benefit from them was greater than the cost, they continued to take the medication. A positive aspect of the biomedical health interventions in the area was that they were run and staffed by locals of the same ethnic group. So, while the community did not trust the government at large, they did trust the health professionals employed on a personal level. However, we also encountered a situation where one individual who had grown up in one of the villages there and came from a modest background, later was perceived as arrogant and driven by financial pursuits by community members after becoming successful. This individual was often used for sensitization by the MoH/government, which backfired as people did not trust in him despite their shared background and family history.

While exploring community perceptions towards MDA and the distribution of Ivermectin, a drug distributed to treat and prevent onchocerciasis, the general impression was that more people were taking IVM now as compared to when it was first introduced. In addition to continuous sensitization, the research showed that people got a sense that the medication helped relieve symptoms and there was a better understanding of the side effects and why they occurred. However, some were still refusing to take the medication. In addition to common fears of side-effects, one reason for refusing up-take was that they did not trust the purpose of the medication. Some households perceived it to be a government strategy to reduce the population in the region. The village health worker who distributes IVM said, “People argue that yes, this government seems to be having a plan to finish off the people of northern Uganda. Why is it that they are bringing these drugs here and not to his (current leader, Museveni’s) village?” (IDI, female, ivermectin distributor)

Consequences for research activities

Due to the spatial and temporal clustering of NS, most studies have been conducted in a limited number of villages since the war ended. The village of Tumangu, for example, in Kitgum district is one of the most popular sites for research because it has had a high number of cases according to health officials. Certain studies reportedly took body samples of children with NS, including blood, skin snips, urine samples, and brains for autopsies. Some children were also reported to have been taken to the U.S.A. for further research. For community members, all this research, perceived as invasive, has not produced concrete results on the aetiology of the disease, which leads to feelings of anxiety, research fatigue and suspicions around the motives of the foreign researchers. The reported lack of dissemination of results lead to bits and pieces of information being shared in the form of gossip and rumours. People further perceived the lack of dissemination as purposefully withheld information and reinforced the belief that foreign researchers were simply using their children’s body parts for profits. Complaints were present across all study villages without exception i.e. researchers came, took information and never returned with results. This has led to research fatigue, distrust and desperation about finding a cure or answer. “I wonder if researchers are just making money with our children’s samples”. (IDI, mother of NS child)

As a consequence of the lack of dissemination in addition to the absence of concrete results, people stated their intention to refuse co-operation in further research. Suspicions about the government’s supposed ill-intentions towards the Acholi were increasing, limited dissemination further reinforced their suspicions of researchers and the government purposefully withholding information to cover up their involvement. Box 3 illustrates how research results were being disseminated as gossip, which created an opportunity for rumours to be spread and alternative interpretations to be mooted. Similarly, inadequately planned dissemination as depicted in Box 4, proved futile and created even more confusion and distrust in the community.

Conclusion and recommendations

Given the ambiguity around NS, and the political context in Northern Uganda, moving forward NS requires the building of trust, the de-politicization of response, and more clarity and transparency. Trust can be built by engaging the community and making them active participants in every stage of the intervention, be it research, service delivery for treatment and prevention, and sensitisation. In addition, community participatory implementation and research methodologies should be used that include continuous community involvement and feedback loops to address rumours and feelings of resentment. This is best achieved through the extended involvement of the community including day-to-day interactions with implementation staff. By creating continuous feedback loops, the sharing of information and engaging in active dialogue, ambiguities around treatment, prevention and the intention of the research will be reduced. Dissemination must be planned. Well-prepared sessions on interim research findings must be conducted at regular intervals despite inconclusive results. This could be a requirement from the Ugandan Ethics Committees and the national Council for Science and Technology for every research, particularly on NS.