Study setting and population

The centre is a national referral facility that provides diagnostics, treatment, and management for people of all ages with leprosy, BU, or tuberculosis. Established in the mid-1920s by American Methodist missionaries, the centre initially provided rehabilitation services for people affected by leprosy, subsequently leading to a colony of people with leprosy in the surrounding area. The centre incorporated tuberculosis care in the early 1990s during the First Liberian Civil War. Based on WHO-driven initiatives to integrate case management for skin NTDs, the Ministry of Health decided to upgrade the centre’s capacity, incorporating BU care with leprosy care since 2015. Following the National Strategic Plan for the Case Management of NTDs, centre staff was trained in laboratory diagnostics and care for BU[19].

Members of the community or health workers refer potential patients affected by BU or leprosy to the centre on the basis of clinical features. Suspected cases are confirmed upon arrival at the centre using acid-fast bacilli (AFB) smears for both diseases. Most patients with BU and leprosy are managed as in-patients and receive long-term care. Both patient groups cohabit and receive care as a single group with shared facilities and health workers. Patients with sufficient mobility might be managed as outpatients if they are able to commute daily for case management activities. Lodging and treatment for patients with tuberculosis occurs in a separate building.

At the centre, patients with multi-bacillary leprosy, defined as having at least 5 skin lesions, receive one year of multidrug therapy (MDT) with rifampicin, clofazimine and dapsone. People with pauci-bacillary leprosy (<5 lesions) receive MDT for six months. People with BU receive 56 days of rifampicin plus clarithromycin or streptomycin, according to availability. Both patient groups receive daily wound care for their lesions which include extensive skin ulcers potentially leading to scarring and contractures in BU and neuropathic ulcers in leprosy [4]. Other skin manifestations of leprosy such as skin patches and nodules do not require wound care. All laboratory procedures, care activities, lodging and food provision are offered free of charge to patients; funding is sourced from a variety of external donors including the German Leprosy and Relief Association (GLRA), and coordinated via the Ministry of Health. During the time of the fieldwork, approximately 90 patients with BU and 30 patients with leprosy were receiving care at the centre.

In-depth interviews

We sought 20 patients (“cases”) with a diagnosis of BU (n = 10) or leprosy (n = 10). We included patients who had received care at the centre for at least two weeks to ensure they had experienced case management activities. We excluded patients with cognitive impairment that could make interviewing difficult, and underage (<18 years) patients without the presence of a guardian since they could not legally provide written informed consent.

During the first week of field work, potential participants were selected by a researcher (MP) during informal conversations or via clinical chart review to identify patients that met the inclusion criteria. We invited patients to participate in the study during delivery of chemotherapy and wound care services, or other activities in common areas in the centre. We purposively sampled participants across the range of ages, genders, languages and counties in the sample to ensure maximum variability of experiences. Health workers did not have a role in selecting potential study participants.

Interviews were semi-structured and focused on eliciting patients’ experiences of illness and health-seeking before admission to the hospital, as well as experiences of case management and rehabilitation after admission. During the first week of fieldwork, preliminary question guidelines were validated with a subset of inpatients (n = 4) not included in the final study dataset. We continued to refine interview guidelines during data collection to include additional probing on emerging themes. Interviews took place in a private office at the centre and lasted between 38 and 60 minutes. All participants were interviewed in English by the first author, with support of translators for local languages (Bere, Gio and Mano). To obtain rich data, interviewing was conducted using respondent-centred approaches, applying interview guidelines flexibly while allowing participants to take control of the conversation and using probing techniques after their answers. All interviews were audio recorded. Information on the gender, age, and diagnosis of cases was extracted from clinical charts.

Field notes

The lead researcher’s observations, perceptions, and experiences of interacting with patients and staff were collected as daily field notes or recorded as voice memos, and transcribed at the end of each day. Unstructured conversations with five staff members (two nurses, one doctor, one laboratory technician and one programme manager) were also included to contextualise the content of in-depth interviews with patients.

Photography

The study involved the production of photographs as primary data. Using an instant camera (Fujifilm Instax Wide), the case management setting, activities and routine procedures, including lesions of patients, were documented during the study period. These photographs did not include patients’ faces or identifying features. Additionally, photographs from atlases of leprosy[20] and BU[21] were used to depict different categories of wounds. During in-depth interviews, both types of photos were integrated into the interview guideline and shown to the study participants as physical prints. Discussions on what they saw, thought, and experienced provided additional meaning to concepts elicited through verbal questions.

Data management and analysis

Audio recordings from all in-depth interviews were transcribed using Microsoft Word 2016 (Version 16.13) with support from a multi-lingual research assistant. English portions of the interviews were transcribed verbatim. Thematic analysis occurred simultaneously with data collection. After six interviews, two clean transcripts were uploaded to Dedoose version 8.0, a software for qualitative analysis. We used a first cycle of coding to develop a tentative codebook and identify emerging themes, using primarily an inductive approach. Deductive coding based on a literature review of patient medical and social experiences of skin NTDs from other settings also contributed to initial codebook development. This codebook served as a provisional start list to develop a case-theme matrix in Microsoft Excel 2016 (Version 16.16.15). The matrix included rows for all cases and columns for each tentative theme, facilitating within-case and across-case analysis. During fieldwork, excerpts from interview transcripts were entered into the matrix in the appropriate case vs. theme cell using pattern coding. Matrix cells also contained descriptive and analytic memos concerning the specific theme in the context of each specific case. The matrix was rearranged and recompleted iteratively throughout the analysis process, as new themes emerged and others converged into larger themes. Thematic saturation for main themes was reached by case 16, four additional cases were included aiming for maximum variability in experiences. Themes were condensed into narrative descriptions of findings. We reported our findings as patient experiences to avoid strictly biomedical interpretations that might further remove future programming from users’ needs. While other approaches, such as emphasising experiences of health workers and operational challenges for programming are also needed, trustworthiness in the exploration of socio-cultural themes such as stigma, disability, and rehabilitation required a patient-centred approach[17]. Representative quotes were embedded within the results to exemplify themes, with minor grammatical alterations to improve readability. Field notes and primary photographs were used to complement descriptions and provide additional context[22]. Case vignettes and descriptions were summarized in Table 1; clinical severity and disability classifications were reported according to WHO guidelines[21].

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Table 1. Case description and vignettes.

https://doi.org/10.1371/journal.pntd.0008030.t001

Ethics

This study was approved by the Ethics Committees of the London School of Hygiene and Tropical Medicine (15584) and the Liberian Ministry of Health (1802088). Those who expressed interest to participate gave their written informed consent. As most participants were illiterate, a research assistant ensured that participants understood the study procedures thoroughly after a verbal explanation in the patients’ mother tongue. Patients who were under 18 years gave verbal informed assent, and were interviewed in the presence of guardians, who gave written informed consent. Participants received a towel, a piece of soap, and a photograph of themselves, equivalent to less than 2.00 USD as compensation for their time. All photographs were taken with the verbal consent of those depicted, including staff, and were shown to them immediately, receiving favourable responses. Due to the stigmatising nature of BU and leprosy, all photographs were produced in the ethical framework of representing vulnerable subjects[23].

“Intertwining” disease identities and inter-disease stigma

Due to its history, the Centre and its surroundings had a negative reputation as a place of leprosy, disability, and amputation. Participants mentioned that members of the centre’s surrounding communities were sometimes labelled as “leprosy people”, which made others scared of approaching the facility. The recent inclusion of BU care within a well-known leprosy rehabilitation facility caused some apprehension among newcomers with BU, who anticipated leprosy-related stigma:

“I was thinking that this place was only for leprosy, not for sores [ulcers] like this […] I said, so when I come, they will not cut [amputate] my foot?” (Case 013, BU, F, 51)

Some participants appeared unable to distinguish between the aetiologies of skin ulcers from the two diseases, or felt distinguishing them was unnecessary. They perceived both conditions to be “intertwined” or related to one another along a continuum of the same disease, suggesting that the integration of care had created new dimensions in old discourses about leprosy:

“Buruli ulcer and leprosy, they are intertwined. […] When the Buruli ulcer, it stays long on you, I think leprosy will join into it. Then your foot and other things will be gone [amputated], so they are intertwined. Because it is sore and caused by bacteria and diseases.” (Case 007, BU, M, 22)

Other participants perceived the diseases as separate conditions but with similar attributes. Leprosy was perceived to be more severe because of an expectation of amputation and social discrimination. Such distinctions helped construct social identities that shaped notions of otherness among patients with the different conditions within shared spaces:

“I have my infection, they have theirs, ok?” (Case 015, MBL, M, 46)

These notions of “them” and “us” shaped inter-disease stigma among both patient groups within the centre, manifested as fear of acquiring the other disease. Some people affected by BU reported “feeling sorry” for people with leprosy, and feared developing leprosy themselves. This sometimes motivated them to use outpatient rather than the integrated inpatient care unit, and made them avoid common bathrooms and mosquito bites in the centre, both thought to facilitate leprosy transmission:

“The reason [I stay home] is that I have Buruli. You have leprosy, then you laying down as a leprosy patient, then I am laying down as a Buruli patient. Mosquito, is able to bite you, and come and bites me. So, the fear is that the person who has leprosy and the person who has ulcer can’t be in one place. I am afraid.” (Case 011, BU, M, 47)

Stigma was bidirectional, as newcomers with leprosy also avoided people in early stages of BU treatment due to the smell of the ulcers and fear of transmission:

“First I was afraid they were going to put me in a room with some of the patients that have the sore. I said to the people: I’m not sleeping there, I’m not gonna get the sore too.” (Case 006, MBL, M, 21)

Health workers, however, pointed out that inter-disease stigma seemed to be buffered over time by morning health education sessions, where information about BU and leprosy transmissibility was frequently discussed. This knowledge was evident among some participants who described leprosy as not transmissible after patients receive medication, and that BU was transmissible by walking in swamps but not from person to person. Morning sessions also facilitated relationship-building among people with similar backgrounds but different diseases thereby encouraging communal living. This was apparent in the case of a man with BU who frequently spent time with a man affected by leprosy. Both young men were from Guinea and were learning English during their time at the centre. After asking for a portrait of them taken together, the man with BU described the photograph as follows:

“This is me. And my Guinea man. Leprosy man. From Guinea. My friend.” (Case 002, BU, M, 22).

Establishing an intertwined patient community helped people from both disease populations to support each other emotionally and instrumentally throughout integrated medical activities, discussed next.

Medication and dressings: Experiences of integrated chemotherapy and wound care

Most participants were aware of differences in the types of medication and wound care required for each disease. Both patient groups experienced receiving daily medication as the cornerstone of their rehabilitation: tablets and pills were described as effective and appreciated for their ability to remove illness and its attached social burden. Medication inspired hope about a future without disease:

"I feel comfortable, when there’s life, there is hope. Because I am going through medication, and you know my time is not yet over." (Case 015, MBL, M, 46)

Though their one year course of medication was perceived as lengthy, people with leprosy framed completing it as necessary to restore their social identities:

“I will say (to others in the community): I took the medicine and I don’t get any sickness now. You must not be afraid of me, you come around me, the way we were joking before, it will be like that now." (Case 009, MBL, F, 66)

A few people with BU experienced only limited wound improvement and maintained positive AFB smears, requiring a second course of treatment with the same regimen. This caused some to doubt the treatment efficacy:

"What I believe in my mind is that BU has no good treatment yet, they have not discovered it for this sickness.” (Case 011, BU, M, 47)

Dressing of skin ulcers was provided simultaneously to all people affected by BU and a minority of those with leprosy. Discussions about this activity emotively described the enacted stigma people experienced associated with smell:

“The way of the sore, and the smell of it, people don’t want to sit around you. Yeah, they think they might get it, they think it’s transferrable.” (Case 006, MBL, M, 21)

Both groups perceived wound cleaning and dressing as healing by taking away germs, replacing foul smelling bandages, and avoiding flies and felt this helped people avoid stigma.

Attitudes towards wound care were also shaped by experiences of pain, which were prominent during the initial stages of wound cleaning. Upon admission, people with skin ulcers underwent manual debriding consisting of removal of necrotic tissue, fluid, and plasters of mud remaining from traditional medicine practices. Some participants interpreted the reduction of pain throughout the wound care process as evidence of improvement, or as a necessary experience to achieve cure, as discussed by a woman with BU while describing a photograph of the wound care room:

“In this place they can change the bandage, and this is the place I said that day that you must take my picture so it can be remembered, because I suffered under this thing [Buruli ulcer] too much […] So I want you to give me the picture when I go, then I can show it to my children, in this place I was cured”. (Case 013, BU, F, 51)

Patients with severe BU required squeezing and draining of subcutaneous accumulations of fluids. Pain management was limited to injection of intramuscular diclofenac, the strongest analgesic available. Instances of screaming, intense agitation, and fainting were observed during fieldwork and documented in participants’ testimonies. Such experiences contributed to occasional refusal or resistance to participate in wound care activities. This was the case of a young man with BU frequently seen arguing with staff during wound cleaning. He described a photograph of patients queueing outside the wound care room as follows:

“The time we can be going for dressing our hearts can be beating, we [are] going [to] feel pain here. Any time we see this place, each time we know our hearts can be disturbed.” (Case 019, BU, M, 17)

People with large wounds due to BU often arrived at the centre presenting with fever, infected wounds in need of antibiotics, and severely malnourished, especially after prolonged and unsuccessful experiences with traditional medicine, as expressed by the mother of a young boy with extensive BU lesions in both legs:

“That was four months we made there [spent at the herbalist], it was not working, he was just reducing the body, (…) losing weight.” (Case 014, BU, M, 15)

Such severe cases required surgical management such as amputation, skin grafting or extensive debridement under anaesthesia, which, theoretically, could be coordinated via referral to a neighbouring hospital. However, staff complained that they were discouraged from organising referrals because of resistance from staff at receiving facilities who feared disease transmission or cited limited resources to cover surgical costs.

Complicated cases such as these thus challenged the centre’s management capacity. The boy severely affected by BU described above died the day after participating in an interview due to sudden onset of diarrhoea and dehydration complicating his vulnerable state. Lack of emergency equipment, overnight medical staff, and capacity to establish a central venous catheter impaired adequate shock management. In the aftermath of the emergency, two health workers admitted that deaths from shock and complications among people severely affected by BU was a recurrent and particularly distressing problem.

“Small, small”: Progressive visible improvement, normalisation and rehabilitation

Notwithstanding instances such as the one described above, favourable experiences of case management dominated participants’ narratives; these emphasised the external visibility of improvements and the reduction of felt stigma and the anxiety caused by anticipated stigma. Testimonies highlighted the noticeable closing of open sores in BU and disappearing of skin marks in leprosy as desirable outcomes of treatment. This was highlighted by a participant describing the experience of a family member who previously received early treatment for leprosy, and whose favourable results included resuming economic productivity and avoiding social isolation due to lack of discrediting scarring:

“A decent man, he’s working! The whole fingers correct, the toes correct, everything on him is fine! No problem, you can’t even know he had leprosy." (Case 006, MBL, M, 21)

Improvements in mobility and decreasing dependence on assistive devices such as wheelchairs, crutches and walking sticks were also emphasised in participant’s narratives. Affected limb movement and function was facilitated by patient’s own motivations to resume social and economic productivity, and by participants’ fear that their bodies would be permanently disabled. Testimonies about the rehabilitation process also acknowledged deficits in the centre’s programming. While assistive devices were available from a local workshop that facilitated makeshift wheelchairs and shoewear, formal physiotherapy services including guidance in rehabilitative exercising were not available due to understaffing. Weekly medical rounds focused on individual adherence to chemotherapy and continuous wound care, and tended to neglect physical rehabilitation plans to prevent lasting functional disability. This had severe consequences for some people affected by BU, for whom a lack of physiotherapy and guidance led to disfigurement potentially incompatible with productivity:

“I can’t come back to my normal stage (…) The way I used to work with my two hands, it will not be like that way now. Maybe sometime, if I don’t get some family to help me, to get some money, to make little business and then sit down… I will not be working hard again, that’s the only thing I am thinking about, the work.” (Case 012, BU, M, 42)

Overall, patients appreciated the long-term commitment to care that the programme and staff provided. Both groups of participants referred to these improvements using the phrase “small-small” to signify how cure occurred slowly, but surely:

“They started to dressing me every morning, give me medicine, they are doing well. At least I can see myself moving on my feet now small, small.” (Case 001, BU, F, 36)

Case management at the centre was described as a rehabilitating and “normalising” experience that addressed both disability and stigma, as exemplified by a man with BU while discussing a photo of a patient waiting area:

"People who come here are people who are getting well […] They [are] able to walk by themselves […] They feel that they are getting normal, they are getting better.” (Case 011, BU, M, 47)

Furthermore, bringing together both patient groups in a single facility for this treatment had the effect of blurring the lines between disease identities, not only shaping unique configurations of inter-disease stigma, but also linking patient groups within a single patient community. This was verbalised by the mother of the boy with BU who died, who described the way integration of care in one facility intertwined disease identities in the collective imaginary:

“The sore people and the leprosy people they are one, they are in one place.” (Case 014, BU, M, 15)

Conclusion

Based on patient experiences, integrating case management of leprosy and BU in this context appears to be effective in addressing the needs of people affected by these diseases. Integration was experienced positively by patients, but reshaped discourses on disease identity around unique inter-disease stigma configurations. Patients experienced consequences of limited resources, such as lack of support to reduce lasting disability, and poor management of BU complications.

Our study further supports the role of applied social sciences in locating people’s experiences at the forefront of programme design and implementation. Future social science research should study how stigma is shaped and performed under the new integration paradigm for the management of skin NTDs. Further patient-centred research and health system strengthening are needed to support the continued development of sustainable and coherent integrated care programmes for these diseases.