Caregiver burden in Buruli ulcer disease: Evidence from Ghana.

BACKGROUND: Buruli ulcer disease (BUD) results in disabilities and deformities in the absence of early medical intervention. The extensive role of caregiving in BUD is widely acknowledged, however, associated caregiver burden is poorly understood. In this paper we assessed the burden which caregivers experience when supporting patients with BUD in Ghana. METHOD/ PRINCIPAL FINDINGS: This qualitative study was conducted in 3 districts in Ghana between August and October 2019. 13 semi-structured interviews were conducted on caregivers of BUD patients in the local language of Twi. Data was translated into English, coded into broad themes, and direct content analysis approach was used to analyse results. The results show the caregivers face financial, psychological and health issues as a consequence of their caregiving role. CONCLUSION/ SIGNIFICANCE: This study found significant caregiver burden on family members. It also highlighted the psychological burden caregivers experience and the limited knowledge of the disease within endemic communities. Further research is needed to quantify the caregiver burden of BUD at different economic levels in order to better understand the impact of possible caregiver interventions on patient outcomes.

Financial burden of health care for Buruli ulcer patients in Nigeria: the patients' perspective.

BACKGROUND: The economic burden of Buruli ulcer for patients has not been well-documented. This study assessed the costs of Buruli ulcer care to patients from the onset of illness to diagnosis and to the end of treatment. METHODS: This was a cross-sectional cost of illness study conducted among patients with Buruli ulcer in four States in Nigeria between July and September 2015. A structured questionnaire was used to collect data on the patients' characteristics, household income and out-of-pocket costs of care. RESULTS: Of 92 patients surveyed, 54 (59%) were older than 15years, 49 (53%) were males, and 86 (93%) resided in a rural area. The median (IQR) direct medical and non-medical cost per patient was US$124 (50-282) and US$3 (3-6); corresponding to 149% and 4% of the patients' median monthly household income, respectively. The overall direct costs per patient was US$135 (58-327), which corresponded to 162% of median monthly household income, with pre-diagnosis costs accounting for 94.8% of the total costs. The direct costs of Buruli ulcer care were catastrophic for 50% of all patients/households - the rates of catastrophic costs for Buruli ulcer care was 66% and 19% for patients belonging to the lowest and highest income quartiles, respectively. CONCLUSIONS: Direct costs of Buruli ulcer diagnosis and treatment are catastrophic to a substantial proportion of patients and their families.

Diagnosis delay and duration of hospitalisation of patients with Buruli ulcer in Nigeria.

BACKGROUND: Delayed diagnosis of Buruli ulcer can worsen clinical presentation of the disease, prolong duration of management, and impose avoidable additional costs on patients and health providers. We investigated the profile, delays in diagnosis, duration of hospitalisation, and associated factors among patients with Buruli ulcer in Nigeria. METHODS: This was a prospective cohort study of patients with Buruli ulcer who were identified from a community-based survey. Data on the patients' clinical profile, delays in diagnosis and duration of hospitalisation were prospectively collected. RESULTS: Of 145 patients notified, 125 (86.2%) were confirmed by one or more laboratory tests (81.4% by PCR). The median age of the patients was 20 years, 88 (60.7%) were >15years old and 85 (58.6%) were females. In addition, 137 (94.5%) were new cases, 119 (82.1%) presented with ulcers and 110 (75.9%) had lower limb lesions. The mean time delay to diagnosis was 50.6 (±101.9) weeks. The mean duration of hospitalisation was 108 (±60) days. Determinants of time delay to diagnosis were higher disease category (p=0.001) and laboratory confirmation of disease (p=0.02). Determinants of longer hospitalisation were; multiple lesions (p=0.035), and having functional limitation at diagnosis and undertaking surgery (p=0.003). CONCLUSIONS: Patients with Buruli ulcer have very long time delays to diagnosis and long hospitalisation during treatment. This calls for early case-finding and improved access to Buruli ulcer services in Nigeria.

Economic inequality caused by feedbacks between poverty and the dynamics of a rare tropical disease: the case of Buruli ulcer in sub-Saharan Africa.

Neglected tropical diseases (NTDs) have received increasing attention in recent years by the global heath community, as they cumulatively constitute substantial burdens of disease as well as barriers for economic development. A number of common tropical diseases such as malaria, hookworm or schistosomiasis have well-documented economic impacts. However, much less is known about the population-level impacts of diseases that are rare but associated with high disability burden, which represent a great number of tropical diseases. Using an individual-based model of Buruli ulcer (BU), we demonstrate that, through feedbacks between health and economic status, such NTDs can have a significant impact on the economic structure of human populations even at low incidence levels. While average wealth is only marginally affected by BU, the economic conditions of certain subpopulations are impacted sufficiently to create changes in measurable population-level inequality. A reduction of the disability burden caused by BU can thus maximize the economic growth of the poorest subpopulations and reduce significantly the economic inequalities introduced by the disease in endemic regions.

Household cost of out-patient treatment of Buruli ulcer in Ghana: a case study of Obom in Ga South Municipality.

BACKGROUND: The economic burden of diseases has become increasingly relevant to policy makers as healthcare expenditure keep rising in the face of limited and competing resources. Buruli ulcer (BU), a neglected but treatable tropical disease caused by Mycobacterium ulcerans, the only known environmental mycobacterium is capable of causing long term disability when left untreated. However, most BU studies have tended to focused on its bacteriology, epidemiology, entomology and other social determinants to the neglect of its economic evaluation. This paper reports estimated the household economic costs of BU and describe the intangible cost suffered by BU patients in an endemic area. METHODS: Retrospective one year cost data was used. A total of 63 confirmed BU cases were randomly sampled for the study. Economic cost and cost burden of BU were estimated. Sensitivity analysis was conducted to test the robustness of the cost estimates. Intangible cost measured stigmatization, pain, functional limitation and social isolation of children. RESULTS: The annual total household economic cost was US$35,915.98, of which about 65% was cost incurred by children with a mean cost of US$521.04. The mean annual household cost was US$570.09. The direct cost was 96% of the total cost. Non-medical cost accounts for about 97% of the direct cost with a mean cost of US$529.27. The mean medical cost was US$18.94. The main cost drivers of the household costs were transportation (78%) and food (12%). Caregivers and adult patients lost a total of 535 productive days seeking care, which gives an indirect cost valued at US$1,378.67 with a mean of US$21.88. A total of 365 school days (about 1 year) were lost by 19 BU patients (mean, 19.2 days). Functional loss and pain were low, and stigma rated moderate. Most children suffering from BU (84%) were socially isolated. CONCLUSION: Household cost burden of out-patient BU ulcer treatment was high. Household cost of BU is therefore essential in the design of its intervention. BU afflicted children experience social isolation.

Combining PCR with microscopy to reduce costs of laboratory diagnosis of Buruli ulcer.

The introduction of antibiotic therapy as first-line treatment of Buruli ulcer underlines the importance of laboratory confirmation of clinical diagnosis. Because smear microscopy has very limited sensitivity, the technically demanding and more expensive IS2404 diagnostic polymerase chain reaction (PCR) has become the main method for confirmation. By optimization of the release of mycobacteria from swab specimen and concentration of bacterial suspensions before smearing, we were able to improve the detection rate of acid-fast bacilli by microscopy after Ziehl-Neelsen staining. Compared with IS2404 PCR, which is the gold standard diagnostic method, the sensitivity and specificity of microscopy with 100 concentrated specimens were 58.4% and 95.7%, respectively. We subsequently evaluated a stepwise laboratory confirmation algorithm with detection of AFB as first-line method and IS2404 PCR performed only with those samples that were negative in microscopic analysis. This stepwise approach reduced unit cost by more than 50% to $5.41, and the total costs were reduced from $917 to $433.

Highly sensitive, operationally simple, cost/time effective detection of the mycolactones from the human pathogen Mycobacterium ulcerans.

A boronate-assisted fluorogenic chemosensor in a solid phase is developed, selectively to detect the mycolactones produced by the human pathogen Mycobacterium ulcerans.

"It is me who endures but my family that suffers": social isolation as a consequence of the household cost burden of Buruli ulcer free of charge hospital treatment.

Despite free of charge biomedical treatment, the cost burden of Buruli ulcer disease (Bu) hospitalisation in Central Cameroon accounts for 25% of households' yearly earnings, surpassing the threshold of 10%, which is generally considered catastrophic for the household economy, and calling into question the sustainability of current Bu programmes. The high non-medical costs and productivity loss for Bu patients and their households make household involvement in the healing process unsustainable. 63% of households cease providing social and financial support for patients as a coping strategy, resulting in the patient's isolation at the hospital. Social isolation itself was cited by in-patients as the principal cause for abandonment of biomedical treatment. These findings demonstrate that further research and investment in Bu are urgently needed to evaluate new intervention strategies that are socially acceptable and appropriate in the local context.