Ethics statement

The research was approved by the Biomedical Research Ethics Committee of the Madagascar Ministry of Public Health (reference number: 032/MSANP/SG/-AGME/CNPV/CERBM) and by the Institutional Review Board of the Antwerp Institute of Tropical Medicine (reference number: 1255/18). Verbal informed consent was obtained for all participants in this study as approved by both ethical review boards. Verbal consent allowed to reduce the formality of the procedure, allowing a more natural flow of discussion. It is important to build trust between researcher and participant and, thus, to ensure the quality of data collection, especially when applying informal techniques such as informal conversations [17]. The research team presented the rationale for their presence in the villages and the information about the work to be carried out, after which consent was sought from communities leaders and participants before data collection began. Confidentiality aspects were discussed in depth and time was taken for potential questions. For minors (under 18), consent from parents was sought and ascent from the minor. We chose the place to have the conversation according to both parents’ and the minors’ preferences. Participants did not receive any form of compensation except for some snacks during focus group discussions. All information that could lead to the identification of respondents or their villages has been removed from this publication.

Study context: the PEOPLE clinical trial

The PEOPLE trial mainly aimed at determining an optimal implementation scenario for prophylaxis in contacts of leprosy cases with the antibiotic rifampicin [18]. The trial implemented door-to-door screening for leprosy by a team of health workers, different types of biological sampling, and distribution of rifampicin to contacts in three different ways depending on the study arm. Prior to implementation, sensitization sessions were conducted in the selected villages by community health workers to convey messages about the door-to-door screening activity and about leprosy in general. All leprosy patients identified were offered free treatment in accordance with the national program protocol (see [19] for more details). This anthropological study was carried out ancillary to the clinical trial to explore implementation bottlenecks. Our qualitative research further explored the socio-cultural factors that influenced the implementation of the clinical trial, including community perceptions of the disease and healthcare utilization.

Study sites and population

This qualitative study was carried out in nine out of the 20 trial fokontany (the administrative subdivision below the commune, equivalent to a village) of three rural communes in the Miandrivazo health district in south-west part of Madagascar. For confidentiality purposes and potential stigmatization concerns, the concrete study villages are not mentioned by name. There are several ethnic groups living in the study sites which can be divided into two groups: those known as native groups, i.e., the Bara and Sakalava ethnic groups; and other groups such as the Merina, Betsileo (from the highlands of Madagascar), Antanosy and Antandroy (from the south), and the Antemoro and Korao (from the south-east).

In Madagascar, the health system follows a pyramidal structure with at the top, the most specialized care being provided in 23 hospitals in nine cities including the capital. The referral system supposes patients get referred from lower and more general care structures available in communes or fokontany to more specialized hospitals in districts or regions. At the lowest level of the pyramid primary health care centers (called CSB) are envisioned as the first point of contact of the population with the formal health system. The healthcare centers, which are mainly run by nurses, are meant to be available in villages (but nearly 66% of villages are more than 5 km from a health center [20]). When we mention health centers in this manuscript, we refer to these types of health structures. In the area of the trial study sites, four health centers exist. For leprosy, it is expected that patients can consult there for diagnosis and receive free treatment and follow-up. However, once treatment is initiated, community health workers residing in fokontany may be in charge of following-up patients, including the provision of their monthly medication. It is worth noting that apart from the treatment of leprosy, the treatment of most other skin conditions, even the common and minor ones, is not free of charge in Madagascar and often unavailable in health centers.

Sampling

We followed a purposive sampling strategy, and proceeded by theoretical and snowball sampling [21,22], with concomitant data collection and analysis. Emerging theories determined the next piece of information to be collected and (key) informants involved. The sampling techniques were used to include different types of informants, including people affected and not affected by leprosy; healthcare providers (community workers, public and private health center staff, clinical trial staff, traditional healers); and local authority figures. Sampling and data collection lasted until a point of saturation was reached, i.e., no new information emerged for theme development.

Data collection

Fieldwork was carried out by a team consisting of a female anthropologist affiliated to the National Leprosy Program of Madagascar and one from the Institute of Tropical Medicine in Antwerp Belgium. In the field, gatekeepers were meticulously selected to facilitate movement through the villages and approach key informants. Frequent field visits, including overnight stays, were part of our approach to building trust with communities and immersion in the study context. During each field visit, the anthropologist(s) spent several days in the villages at each of the rural study sites. These data-gathering periods were interspersed with intermittent analyses to guide the further sampling and themes to be explored in greater depth. Semi-structured and in-depth interviews (IDI), group discussions (GD) and participant observation (PO) including informal conversations (IC) were carried out intermittently between September 2019 and December 2020. The interviews were conducted in Malagasy and sometimes in French when addressing the local medical study team, health workers or teachers in the village. Through interviews with key-informants among political, administrative, religious, and traditional authorities, we were able to select gatekeepers that were residents of the villages, had a good reputation and were well known and trusted by community members. We usually conducted PO, including informal conversations, at the beginning of the data collection to gain main ideas about the topics, to select potential informants for in-depth interviews through an IDI or GD and when sensitive topics were intended to be broached or when participants refused an official interview.

Analysis

The PASS-model [23], a model founded on theory to guide the formulation of questions on health seeking behavior and access to care, helped in structuring initial question guides, thematic analysis and data interpretation. The analysis was carried out in two phases during the study. First, analysis was intermittent between the data collection periods (cf. theoretical sampling explained above), followed by internal discussion between team members to guide the next steps of data collection and go into greater analytic depth. Secondly, raw data was processed in its textual form, translated in French (for the data collected in Malagasy) and thematically coded to generate analytical categories using Dedoose qualitative data processing software [24]. The coded excerpts were then compared and interpreted to identify emerging themes and summarize findings.

Study participants

We conducted 66 in-depth interviews; 10 group discussions; 41 informal conversations and 56 observations. See Table 1. Participants were men and women from different age categories: children, young people, young adults, adults, elderly people. Of the 20 patients included in our data, six were enrolled as patients in the PEOPLE trial. In most cases, we obtained data directly from patients (18 individuals), but sometimes from family members (two individuals). The main socio-economic activities of the informants included farming and fishing, with other varieties such as healthcare work (biomedical and traditional), business (trading, selling), handicrafts making, brick making, teaching, driving and panning for gold in rivers. There were other participants of various social statuses: fokontany chiefs, village committee chiefs and community workers.

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Table 1. Distribution of participants by individual characteristics*.

https://doi.org/10.1371/journal.pntd.0013212.t001

(Non) perceived signs and severity of leprosy

Leprosy was commonly referred to as habokana (the fact of having leprosy). The illness was identified by community members when the signs suggestive of leprosy were visible: sores and ulcers on the skin, amputations of limbs, facial deformities, and sometimes reactions with neurological or systemic complications. Therefore, leprosy was also known in the community as manapatapaka, meaning the disease that amputates; and, by older participants as aretin-koditra, i.e., skin disease.

Some patients diagnosed in the early stages of their disease with only skin patches stated they were surprised when receiving the diagnosis, indicating that less pronounced/visible signs were not always interpreted as leprosy.

Skin patches could be embarrassing and even be perceived as a sign of disease, but not necessarily leprosy. The wife of a leprosy patient who lived in a village with a health center said that she had noticed the (insensitive) skin patch on her husband’s knee, the sign that led to his diagnosis for leprosy by the team of the project, for about nine years without this prompting a search for care. “On his knee, there is a patch that doesn’t itch, is not painful but it was there since I saw it the first time when we got married” (Female participant, adult, shopkeeper, IDI).

Moreover, skin patches were often perceived to be caused by frequent contact with river or lake water, common in children and healing spontaneously in adulthood: “We don’t know how, but the spots disappear in adulthood” (Female participant, adult, farmer, GD). These skin problems were interpreted as non-severe and therefore did not lead to an active recourse to care. This perception is in contrast with the representation of leprosy, which is perceived as dangerous, and associated with severe, advanced signs of the disease.

The less visible neurological complications such as paresthesia, or weakness of a limb, and even clawed fingers, were also not clearly identified as signs of leprosy. When these signs appeared, other explanations were considered more likely causes of the symptoms than leprosy, such as long-lasting wounds, curses cast by adversaries, stings from fish fins, etc.

Perceived aetiology of leprosy

The presence of skin patches did not automatically prompt people to seek care. Certain illnesses were perceived to fall within the domain of the healer’s capacity to treat, while other illnesses, such as suspected malaria or stomach-ache, should be taken to a medical doctor. “There are different categories of illnesses [...] When your illness is one that should be treated at the healer, but you take it to the doctor, you won’t be cured.” (Male patient, adult, fisher and fishnet maker, IDI). In case of perceived signs of leprosy, their aetiology was primarily considered mystical in nature.

Perceived transmission of leprosy

Some informants reported that leprosy is transmitted from one generation to the next. This perception was also accepted by patients where the illness clustered in the family and contributed to the gravity associated to the disease: “The doctors say it’s transmissible, but what scares me is that it could be transmitted to my child” (Male patient, adult, farmer, IDI). However, patients that were the only cases in their family often expressed that the disease was not transmissible. A patient said: “My disease is not transmissible. If it was transmissible, my kids would all have been infected by now” (Female patient, adult, farmer, IDI). People without leprosy often reported to prevent transmission by avoiding contact with patients. Risk for transmission was seen when having close contact (mifanosonosona), bathing in the same water, eating or sleeping with a patient or touching their personal effects such as clothes or eating utensils, touching a sick person’s corpse on the leaking parts of his or her body.

Treatment options for leprosy

Factors driving choice of care for leprosy

Factors driving treatment adherence

Social impact of stigmatization

Leprosy’s perceived association with theft and other moral transgressions, combined with contagion fears, rendered it shameful and stigmatizing. This lead some patients to seek care far away from their village to avoid stigmatization. Although community members reported attenuated stigma over time, manifestations persisted and varied contextually. Some villages excluded patients from community life and households, with patients occupying separate annexed rooms despite single-room family dwellings being typical, or eating separately with dedicated utensils while families shared meals: “If they’ve set aside my plate, it’s because people are afraid of it [the disease]. But if they weren’t afraid of it, they wouldn’t put my plate aside. If I cook separately, it’s because people are afraid” (Male patient, adult, wood seller, IDI).

Informants indicated that a divorce could ensue in the event of a diagnosis of leprosy, and that marriages to patients or their family members can be avoided or cancelled on ground of the diagnosis. The reason for this social distance remained the fear of transmission. Despite a few patients where we observed or were told that social relationships were (self-)limited due to the illness, in many cases, the patients in our study were living with their families, were married and/or had children, and mentioned having family, colleagues and/or friends with whom they had regular contact.

Regarding relationships with healthcare staff, some patients feared village screening visits by medical teams might exacerbate existing stigma. This was linked by some patients and their families to the fact that on the one hand, community health workers and doctors talked about the disease by saying that it is transmissible through contact, which accentuated stigma. On the other hand, a lack of trust in health workers would also have arisen since some patients suspected healthcare workers of disclosing their illness. “We’re stunned, it’s the doctors who are behind this [disclosing our illnesses], they said this thing [my disease] is transmissible. […] Nobody knew about my illness, but it was the people who examined me who spread the word […].” (Male patient, adult, farmer, IDI)

Biomedical definitions of leprosy at odds with local socio-cultural interpretations of leprosy symptoms

Seeking timely care based on the recognition of the warning signs of leprosy (i.e., passive case detection) constitutes the main programmatic approach for preventing invalidities and further transmission in the community. However, in Madagascar leprosy patients are often seen in the formal health system only at later stages of disease progression, namely with visible deformities and complications with neuritis. Our research demonstrates that community perceptions of leprosy do not include early warning signs such as skin patches and milder symptoms of leprosy, impeding timely seeking of a diagnosis and treatment. Indeed, early-stage disease and milder symptoms of leprosy are often not interpreted as leprosy, nor any other severe disease, and consequently do not lead to seeking care.

In this study in Madagascar, the perception of leprosy is linked to immoral and antisocial behavior which can have effects on stigma and access to care, and contribute to the notion that leprosy is incurable according to some community members. A study of a community in Nigeria showed that leprosy is associated with dirtiness and immoral behavior and is therefore considered as a dishonorable disease [25]. This perception accentuated stigma and further limited patients’ access to care in health centers.

Curing leprosy is not as obvious as in other diseases, since it can lead to irreversible disabilities. Even long after treatment, leprosy reactions can occur, which throw cured patients back into a state of serious illness, and lead to visible and irreparable damage if left untreated. In the past, when the disease was not yet treatable, the image that prevailed was that of patients who were isolated from the community to prevent transmission, and who carried disfiguring disabilities until their death. Decades later, this image has not yet disappeared from the collective representation, especially for older participants. Madagascar is, however, no exception. In many places of the world where leprosy has existed, including Europe, perceptions of leprosy have remained associated to a disfiguring life-long condition. In Japan, discriminatory laws persisted until 1997 [26] and (official) isolation, sterilization and abortion continued despite widespread MDT [27,28], reflecting deeply rooted historical perceptions [29–31].

Bridging the gap between the biomedical and traditional care systems

According to our results, patients were more likely to first seek treatment from traditional healthcare providers than formal (biomedical) healthcare providers for perceived leprosy signs, frequently already advanced. In the case of symptoms that were interpreted as more serious, patients would sometimes visit villages several hours’ walk away from their residence in search of a good healer, and answers about their illness. Distance, although a real difficulty, was not therefore a major bottleneck in achieving access to healthcare for leprosy, insofar as patients were able to overcome it when feeling the need to seek care. Depending on the results of the healers’ visits and often with the onset of complications, the patient would turn to biomedical care as a last resort. While the focus of our study was on leprosy, our findings on health seeking practices are likely to apply to other health issues as they are embedded in broader logics and perceptions of health and illness, and in similar challenges relating to access to care.

Several studies in Asia and Africa show that for leprosy [10,13,14,32] (as with other health issues, see, e.g., [33–35]), traditional healers are the first option of seeking care before attending biomedical healthcare facilities. Opting to first consult with healers is suggested to be a factor of case detection delay [10,13,14]. Integrating or collaborating with healers in formal healthcare has been practiced in many contexts and health topics [33–35]. Involving them as social workers to refer patients for timely consultation in health centers could be a strategy to induce both early detection [36] and adherence to treatment. In a study in Indonesia, healers’ activities were valorized as they received training on leprosy diagnosis and treatment using a leprosy booklet helping them during consultations, and then their practices were evaluated [37]. According to a study in Benin, multiple visits to a healer constituted a factor in delaying treatment and resulted in patients being seen at an advanced stage of the disease, yet health workers’ lack of skills in recognizing the early signs of leprosy was also a factor in the high proportion of patients with grade 2 disability [12]. These findings are consistent with those of our study and highlight the need to consider and empower all stakeholders who might be involved in leprosy control for an integrated approach.

For the communities in the study sites, lack of awareness of the possibility of free treatment was also a barrier to obtaining biomedical care. In addition, sometimes, the diagnosis of leprosy would not be made at the first visit to a health center, and in some cases, patients had to consult two or three care providers before receiving a diagnosis and appropriate treatment. This highlights the lack of knowledge among some health workers about leprosy, but also the unsatisfactory welcoming of patients by health workers.

A delay in diagnosis has also been mentioned in other studies, such as in China for instance, where patients could only be treated after a delay of up to 202 months and after visits to different health centers with different specialties [38]. Other presented findings resonate with additional work conducted in different settings. For instance, in a study carried out in Ethiopia, it was revealed that knowledge of leprosy is lacking for patients and leads to a delay in diagnosis and treatment [32]. According to another study in three states of India, when questioned about the reasons that prevented them from seeking care, the patients mainly mentioned that they did not know that these skin patches corresponded to an illness, or they thought that it could disappear spontaneously. Only a small proportion of the study population mentioned not having the financial means to access a health center and four to 51% did not know of a health center where they could go for treatment. [39].

Overcoming (fear of) stigma and increasing awareness of leprosy as a biomedical condition

The representation of leprosy transmission as being hereditary and inter-human, and the perception that it can result from a punishment, fed the stigmatization and led some patients to deny and hide their disease. Avoiding biomedical care was a way not to reveal their illness. Some patients moreover mistrusted healthcare workers to maintain confidentiality about their illness.

Our findings converge with those from Feenstra et al. in Bangladesh, which illustrate the dilemma that patients may face in such a situation between the necessity to disclose the diagnosis as a way to raise awareness and protect family members from infection, and the fear of stigma that could result from being identified as affected by leprosy [40].

Our study additionally highlights the need to improve general awareness about leprosy, both in terms of the content of health education messages and of the way they are delivered, since knowledge about leprosy, its transmission patterns and treatment options was not optimal, and stigma still manifested in various ways. While intensifying awareness by increasing the number of districts to be targeted for leprosy sensitization coupled with the reiteration of sensitization within these communities, would undoubtedly constitute a commendable approach, a crucial and cost-effective recommendation would be the re-evaluation of the content of awareness-raising messages. This should incorporate the early signs of the disease to promote early diagnosis, and caution against the risk of severe outcomes if left untreated, in addition to the fact that from the day a leprosy patient starts treatment, infectiousness is almost zero [41]. In recent years, the national program has started using visual aids depicting skin patches as early signs of leprosy and this initiative seems to start producing positive results. Additionally, our study shows that patients need to be provided with a greater amount of information about (free) treatment options and providers, and the need to be informed about leprosy diagnosis in a socially acceptable way. Health workers still found it difficult to announce the disease to patients at the time of diagnosis and sometimes concealed certain information so as not to shock the patient. The expressions used to disclose the diagnosis played a role in patient’s acceptance of the condition and adherence to treatment. Prior research has underscored the importance of exercising caution when discussing sensitive topics, such as leprosy, within the context of a therapeutic relationship. [42]. Importantly, the care provided for those suffering from leprosy must be humane and contextualized in order to circumvent the exacerbation of stigma [43,44] and to facilitate the culturally appropriate utilization of biomedical treatments [45].

Situating health seeking within a broader systemic perspective

Nevertheless, “looking at access to health care through an isolated ‘stigma’ lens can decontextualize the problem and lead to ineffective solutions”[46] and it remains important to consider all factors determining access to care and health seeking behavior beyond stigma and socio-cultural factors. In a country like Madagascar, active case detection is limited due to lack of logistical and financial resources at disposal of the National Leprosy Program (NLP), particularly in relation to the size and spread of the affected areas.

Several efforts have been made by the NLP and stakeholders to tackle leprosy, such as the introduction in 2015 of active case finding in some health centers combined with sensitization and patient care for leprosy and other common skin diseases [5,47]. During the PEOPLE study, very few refusals (0,1%) were reported for door-to-door screening. In terms of cases detected, a high prevalence of 35 per 10 000 was recorded during the first year of screening, followed by a low incidence rate of 0,57 per 10 000 in the subsequent two years [48]. This suggests that transmission is not rampant in our study context, yet passive case finding implies that a substantial number of cases go unnoticed. The study allowed for an increase in knowledge of health workers in the four district health centers and of community workers in the study sites. It has also enabled regular awareness-raising, active screening of patients and close monitoring of patients during their treatment, even if only for a limited number of years and in a restricted area.

However, while the fight against leprosy faces disease-specific challenges, it is also substantially constrained due to an underfunded, challenged and poor functioning health system more broadly, which remains very difficult to access in many rural parts of the country. As long as there is not an integrated approach allowing for people affected by different (skin) conditions to receive proper diagnosis and affordable treatment, even for simple dermatoses which is not currently the case, it will be difficult for people with minor symptoms, including early symptoms of leprosy, to consider the formal health system as a first option of care.

Strengths and limitations of the study

To the best of our knowledge, this is the first scientific publication to describe community perceptions of leprosy in (one part of) Madagascar.

While we spoke with traditional healers, we were not able to observe consultations with traditional healers. Future studies could include such an observation component to better understand why people go to traditional healers and test an intervention engaging traditional healers to better orientate patients to the health centers when needed, as has been tried elsewhere.

This study was a qualitative study carried out in a limited number of places in one district of Madagascar, and therefore only discusses the context for the studied communities in this district. Given the island’s surface area and the fact that it is home to 18 ethnic groups, each with their own traditions and customs, leprosy perceptions may vary from one place to another. The results cannot be generalized.