Introduction

Buruli ulcer disease (BUD) is a necrotizing skin disease caused by Mycobacterium ulcerans and is recognised by the World Health Organization (WHO) as a Neglected Tropical Disease (NTD) [1]. The disease occurs in more than 30 countries worldwide with the majority of cases in rural parts of West African countries including Ghana [2]. Sporadic cases are also reported in many locations within the Americas, Japan and the West Pacific regions [3]. Buruli ulcer presents initially as a painless subcutaneous nodule, plaque or edema which may progress with time into ulcers with necrotic bases and undermined edges [2]. This results in large disfiguring skin ulcers mainly in children aged 5 to 15 years though all age groups can be affected [4]. The establishment of the WHO Buruli Ulcer Initiative in 1998 together with improved diagnosis and management have led to major gains in understanding disease mechanism, however, its mode of transmission remains an enigma [5].

Prior to the use of antibiotics, surgery was the most effective means of treatment. Currently, the standard of treatment is the administration of oral antibiotics (specifically rifampicin and clarithromycin) daily for 8 weeks [6–8]. Additionally, adjunctive therapies like wound care, debridement and physiotherapy can reduce complications especially when lesions develop close to a joint [9]. Early complete antimicrobial administration gives excellent treatment outcomes, with return to quality of life [2]. On the other hand, when medical intervention is delayed or incomplete, permanent functional disabilities and their associated stigma [10] and psychological burden ensues [11–13]. Exacerbating these issues, an important loss of economic productivity often ensues [14–16]; in one Ghanaian study, this amounted to 265 working days lost due to BUD [17].

Nevertheless, BUD is prevalent in rural communities where there is poor access to health care facilities [18]. Affected individuals have to travel long distances to access healthcare. Although cost of BUD treatment in most endemic African countries is free, supplementary expenses such as costs of feeding, transportation, and prolonged hospitalization are a huge economic burden [19–22]. This creates a barrier for patients to seek early medical intervention, thus leading to worsening disfiguration and disability.

The debilitating nature of BUD, the young median age of disease occurrence, the significant financial burden and frequent hospital visits leads to a wide-ranging role for caregivers such as relatives or loved ones. Per the WHO report on ageing and health, ‘a caregiver provides care and support to someone else; such support may include: helping with self-care, household tasks, mobility, social participation and meaningful activities; offering information, advice and emotional support, as well as engaging in advocacy, providing support for decision making and peer support, and helping with advance care planning; offering respite services; and engaging in activities to foster intrinsic capacity’[23]. Whereas caregiving can strengthen family ties, the role also demands committing significant amount of time and energy for long periods and performing tasks that may be physically, emotionally, financially, spiritually and socially daunting. The extent to which caregivers perceive these tasks as challenging can be termed ‘caregiver burden’[24]; these two definitions are maintained throughout the study. In a previous study, household cost of caregivers of BUD patients was 8.6 times higher than in those that socially isolated themselves from their BUD affected relatives [25].

A number of studies have documented the impact of caregiver burden on caregivers of patients with chronic skin NTDs namely podoconiosis [24], Lymphatic filariasis [26] and cutaneous leishmaniasis [27,28]. In addition, there has been one qualitative study of BUD caregivers to date, conducted in Benin [29]. In this paper, we for the first time, explored the caregiver burden in persons who provide such support to BUD affected patients in Ghana.

Methods

Results

Characteristics of study participants

The demographic characteristics of study participants are presented in Table 1. Of the 13 participants, there were 10 females and 3 males; 8 of the participants were above 35 years. All but one (12 of 13) of the participants were caregivers of former BUD patients whose lesions had healed completely with no functional impairments. One participant was a caregiver to a BUD patient with active category I lesion on the hand. With the exception of one participant who was unemployed, 10 were farmers, 1 was seamstress/farmer and 1 was a trader. Most (10/13) participants were married but 2 were widowed. Most of the caregivers were parents (7 were mothers and 3 were fathers) of the BUD patients. An equal number of participants (4/13) had either primary or secondary education; 5 participants did not have any form of education.

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Table 1. Socio-demographic characteristics of caregivers of patients with BUD.

https://doi.org/10.1371/journal.pntd.0009454.t001

Discussion

While the psychological impact of BUD on patients has become increasingly recognised [11–13], this is the first qualitative study of caregiver burden in BUD in Ghana, and expands a small but important evidence base for this neglected aspect of BUD. The present study also represents one of only a few studies to look at caregiver burden for any NTD [24,26–29].

Caregiver burden, defined in this study as ‘the extent to which caregivers perceive that caregiving has had an adverse effect on their emotional, social, financial, physical, and spiritual functioning [24], was prominent among caregivers of those affected by BUD in three districts in Ghana. We categorized the caregiver burden into four main themes: 1) Personal burden; 2) Impact on other family members; 3) Barriers to accessing care; 4) Support for caregivers. Within these four themes, we have highlighted the significant financial impact of caring for patients with BUD. The loss of productivity an affected BUD patient faces inevitably places significant pressure on the caregiver to provide for the rest of the family. This is reflected by the finding that almost all of the caregivers in this study (12/13) were employed. Financial burden reported here is in keeping with a previous Ghanaian study where the household costs of caregivers of BUD patients rose by up to 8.6 times [25]. Study participants also reported caregiver burden in terms of reduced occupational output, psychological and health issues. In a study on podoconiosis patients’ caregivers in Ethiopia, caregivers provided care for an average of 14.8 and 15.5 days per month for patients with mild/moderate and severe disease respectively. Furthermore, the hardship experienced by family units was not only due to the patients decreased working hours, but, was also due to caregivers’ lack of productivity while providing care [32]. Psychological burden reported by caregivers was directly related to the financial pressure they came under as worrying thoughts of not being able to provide for other family members was troubling and corroborated the study on caregiver burden in another disabling skin NTD, podoconiosis [24]. However, in addition to this psychological impact, caregivers also reported the provision of physical care had a negative impact on their own health and well-being. The amount of time dedicated to providing physical care and the extra responsibilities a caregiver needed to assume placed significant stress on the individual, affecting their health. It was also apparent from our study that like BUD patients, caregivers also faced the issue of stigma in their communities. Stigma in BUD is caused by the presence of large disfiguring ulcers coupled with, an unknown mode of disease transmission, leading to associations with cultural and religious beliefs [10,14]. As a result, although patients do still wish to interact within their communities, affected individuals and their families become socially isolated [33,34].

In BUD management, seeking early complete medical intervention lowers the risk of permanent functional limitations and its associated impact on quality of life [2] and psychological co-morbidities [11,12]. Caregivers narrated how finances and non- availability of simple logistic support became barriers to accessing healthcare for their sick relatives. BUD is endemic in rural communities which usually have poor roads and limited geographic access to major health centres. It is therefore unsurprising that caregivers experienced difficulty with transport, leading to delays in seeking early patient care as reported by one caregiver in our study. One solution to the issue of transportation on caregiver burden has recently been highlighted in work surrounding decentralised BUD patient care [35]. This alternative approach has particular benefits to female caregivers, who have been shown to prefer this approach as it facilitates their frequent role in mobilising caregiver resources for BUD patients in Benin [29].

Support available to caregivers greatly influenced the extent a caregiver felt burdened having to cater for sick relatives. The more support a caregiver received, the less likely the person becomes burdened with his role [36]. Some caregivers reported that they received financial support from health service facilities, friends and family. BUD typically affects people of lower socio-economic status and the high cost of management is financially draining on the patient and his family. In Ghana, antibiotics for BUD treatment are distributed for free under the auspices of the National Buruli Ulcer Control Programme with support from the WHO; but patients and caregivers may incur some cost related to care. Hospital care costs may include direct costs like admission fees (when needed as may occur if sepsis develops from secondary wound infection), wound dressings, fees for surgical procedures like debridement and skin grafting (when done), feeding and indirect costs related to loss of economic activity. Most BUD care provided in Ghana is on outpatient basis but patients and caregivers may still have to cater for transportation to and from health facilities, feeding and other ancillary costs. Other caregivers reported that they did not receive any form of support and this greatly affected them.

The Sustainable Development Goals (SDG) target 5.4 [37] seeks to recognise and value un/under paid care and domestic work. Caregivers of patients with chronic NTDs provide unpaid care. The BUD control programme in Ghana should ensure that there are pathways to provide advocacy for support of caregivers of persons with chronic NTDs like BUD. Caregivers should be included in the programmatic planning for management of patients with chronic NTDs like BUD. National BU control programmes should include packages like education and psychosocial support services that will help improve the experience of caregivers of BUD patients.

While this study has made some novel and valuable findings, there were a number of limitations. The study did not include any children who were caregivers. In Ghana, children may provide such support as running errands, helping with farming and cooking among other household chores. Since BUD disease can affect all ages, it would have been interesting to understand caregiver burden from the view point of child caregivers who are offering such support to parents or other older relatives affected by BUD. There was no provision for field notes and documentations of observations made during qualitative interviews. Although we observed and acknowledged some emotions of participants during the interviews, we did not document any of these. Field notes and documentations are subjective measure assessments and not a substitute for the tool employed. No caregivers of chronically disabled BUD patients were included in the study, thus we were unable to assess the impact on caregivers of lasting functional impairment. Furthermore, the assessed caregiver burden could have been impacted by recall bias as most participants were caregivers of former patients. In spite of these limitations, this study has provided valuable information to allow for a more comprehensive approach to managing BUD patients and their caregivers.

Conclusion

Overall, the debilitating impact of BUD negatively affects not only patients but also their caregivers to a significant degree. Within the context of NTDs, the evidence base for caregiver burden remains restricted to a small number of chronic stigmatising skin NTDs. Therefore, more qualitative and quantitative studies are needed to grow the evidence base in this area. Efforts should be made to provide financial assistance such as waivers on hospital care costs and other incentives for caregivers of BUD patients as this could go a long way to ease the burden of caregiving for BUD in Ghana. Lastly, our study highlights importance of further work on the benefits of integrating psychosocial interventions in NTD management for both patients and their caregivers alike.

Supporting information

Acknowledgments

We are grateful to staff of the network of clinics involved in the provision of care for patients with Buruli ulcer disease (BUD) for their assistance. We thank all the caregivers of BUD patients who participated in the study; we appreciate their time and effort in providing care for patients. We thank Jonathan Adjei, a researcher at the Kumasi Centre for Collaborative Research for his assistance with the study.