The World Health Organization (WHO) Roadmap to End Neglected Tropical Diseases (NTD) includes the elimination of lymphatic filariasis (LF) as a public health problem in at least 58 of the 72 LF-endemic countries by 2030. Elimination of LF as a public health problem is defined as 1) an infection rate sustained below transmission thresholds, and 2) the availability of an essential package of care for people with existing clinical disease (1). The clinical manifestations of LF are hydrocele and lymphedema, and these conditions are exacerbated by secondary infections causing acute dermato-lymphangitis (ADL). Disability from LF affects 42 million people resulting in an estimated loss of 5.25 million disability adjusted life years (DALY) (2). Hydrocelectomy surgery offers a permanent cure for people affected by hydrocele, but lymphedema cannot be cured and treatment entails lifelong self-management by affected persons with the support of their family and broader community (3). The WHO recommends similar self-management interventions for people affected by podoconiosis (4), which causes lower limb lymphedema after bare-foot exposure to irritant soils (5). Together LF- and podoconiosis-related lymphedema cause pain, disfigurement, disability, and social exclusion for 20 million people globally (6, 7).

The extreme suffering of people affected by hydrocele and lymphedema was the impetus for establishing the Global Program to Eliminate LF (GPELF), which sets out validation targets for the implementation of 1) mass drug administration (MDA) of preventive chemotherapy, and 2) morbidity management and disability prevention (MMDP) services for people with existing disease (8). Since the inception of the GPELF in 2000, targets for MMDP have lagged behind MDA (9) and by 2019, 62 endemic countries reported that MDA had been scaled up to all endemic areas or completed, but only 41 countries reported availability of the essential package of care in any endemic areas (10). The Roadmap targets a 75% reduction in DALYs by 2030 (1) and a significant acceleration of interventions to reduce and prevent morbidity and disability will be needed in order to achieve this.

A distinct feature of the Roadmap is greater ownership by national and local governments, including communities. Achieving this in LF- and podoconiosis-endemic areas will depend on educating all affected persons in evidence-based and effective lymphedema self-care, and on providing support to perform the recommended self-care activities. Currently, WHO recommendations include education on the causes of lymphedema, a daily regime of washing and drying the affected body parts, meticulous skin care to prevent secondary infections (ADL), and elevation and exercises to promote lymphatic circulation (8). There have been multiple studies and two systematic reviews which show the benefits of these core self-care practices for the individual (11, 12), and the economic gains to the whole community (13). Implementation of lymphedema-management at this level is only sustainable if it addresses any challenges and barriers to performing the daily self-care regime, and these may be specific to country, region or individual. Here, we report on the results of patient, caregiver and local health worker questionnaires, and patient journals collected during a study on an enhanced self-care protocol (14) for people affected by moderate and severe lymphedema in Bangladesh (15) and Ethiopia (16). Insights from these diverse populations may be transferable to other LF- and podoconiosis-endemic counties and contribute to achieving the overarching goal to leave no-one behind.

Patients in both countries experienced significant improvement in objective measures of lymphedema status and these results have been published for each country (15, 16).

The WHO Roadmap to end NTDs obliges us to measure the public health impact of interventions (1). However, countries seeking validation in the GPELF need only measure actions such as mapping cases, and reporting the number of health centers capable of offering services (8). There is no requirement to report on the number of people who are trained in lymphedema self-management, or the frequency of their self-care practice (8). However, results presented here from both Bangladesh and Ethiopia showed that while most health workers had good knowledge on lymphedema causes (>90%) and the need for limb hygiene (>70%), knowledge among the end-users, the patients and their caregivers was much lower, less than 25% for either population. This knowledge gap must be addressed, and the goal to reduce the suffering of people affected by lymphedema requires that effective tools are placed in the hands of those affected and their families (23). New approaches and tools are being developed to help health workers objectively assess the patient’s condition (24–26), but without measures in the validation dossier on meaningful actions taken by the end user in terms of quality-of-life, improvement in productivity, or reduction in disability, we fail to capture the public health impact of lymphedema interventions.

Chronic skin disease from LF, podoconiosis and other NTDs are managed by similar practices and present considerable potential for cross-cutting service delivery in an integrated health management system (4). To realize this, general and community specific challenges and barriers must be considered, and in our study contrasts in topography and climate provided a backdrop to knowledge and behavior in each community. In Bangladesh where water is plentiful and communities are closely clustered around low-lying rice paddies and vegetable gardens, people washed more frequently, performed more leg exercises but walked less. In Ethiopia where villages are separated by large tracks of dry farmland and water is scarce, people washed less frequently and walked more, doing fewer of the leg exercises. Elevation was used the least in both countries, which is consistent with other reports from Ethiopia (27), Zambia (28), Sri Lanka (29), and in Haiti (30) where compliance with leg washing was 88% while less than 50% of the study cohort were sleeping with the foot of the bed raised. We found several patient specific factors associated with adherence in each country but few previous reports for comparison. Age, sex and literacy have been discussed in regard to lymphedema knowledge and health seeking behaviors (31, 32), or in association with lymphedema stage or ADL frequency (23). Addiss etal. (33) reported that overall adherence to lymphedema self-care practices was significantly associated with being female (OR 1.6, 95% Confidence Interval [CI] 1.0–2.5, P = 0.05) and over 40 years of age (OR 1.5, 95% CI 1.1– 2.0, P = 0.02), but no other studies have reported on factors associated with performance of individual self-care activities. Adapting which practices are recommended or emphasized to accommodate environmental, personal and cultural factors may increase the potential for widespread acceptance and integration of beneficial practices across the whole community.

Data on adherence to self-care practices is typically collected either by survey in which participants are asked to recall events retrospectively, or via a patient journal where activities are recorded on a daily or weekly basis. In our study we collected adherence data using both methods with general concordance in the results on performance of the self-care activities, suggesting either method may be suitable for assessing adherence. However, in both countries, more acute symptoms were reported in the journals than via the surveys, which indicates that people may have experienced ADL symptoms more frequently during the study period than were captured by recall at the end. Likewise, more days where the patient was unable to work due to lymphedema appeared in the journals than were recalled by survey. It is our view that a daily journal or chart kept by the patient and/or their caregiver provides more accurate data on theses markers of disease than retrospective questionnaires.

Unfortunately, we did not ask patients or caregivers if they had received prior MDA or training in lymphedema self-management and data on this may have helped to elucidate the disconnect between knowledge held at the health center and general awareness found among patients and caregivers at baseline. Furthermore, in each country some of the original caregivers were replaced by a new caregiver during the course of the study. While we accounted for this in our before and after comparison on caregiver knowledge, it should be noted that repeated training of new stakeholders will be needed. The original study design and sample size calculations were based on the primary outcomes in the RCT and the results are limited by the lack of longer term follow up. A further shortcoming was in only extracting data from the first week of each 4-week journal to calculate the weekly adherence scores, however it was noted that the first page of each journal generally had the most complete entries with blank spaces and skipped pages more frequently among later pages. The study strengths were in the inclusion of a large number of participants in two contrasting settings, and the collection of data on caregivers and health workers as well as patients. We anticipate these observations on knowledge and adherence in Bangladesh and Ethiopia will be useful to other endemic countries with similar populations.

It is well documented that people affected by lymphedema at all stages have impaired qualify of life and increased financial pressures (34), and that introduction of a hygiene centered self-care program can improve symptoms and reduce ADL episodes (11, 12). In our study, people affected by moderate and severe lymphedema and their caregivers were able to learn and apply lymphedema self-care with a high level of adherence over 24-weeks. Such community based and patient-oriented health services are fundamental to achieving the Sustainable Development Goals (SDG) in universal health coverage, and for achieving global targets for action on NTDs (SDG 3), alleviating poverty (SDG 1) and hunger (SDG 2), enabling people to pursue an education (SDG 4) and lead productive working lives (SDG 8) (1), and to meet the core Roadmap agenda to move away from external drivers and enable in-country autonomy in program delivery (1).

In Bangladesh and Ethiopia, lymphedema knowledge and self-care practice increased significantly 24-weeks after lymphedema self-care training. Daily performance of lymphedema self-care can improve clinical outcomes and social inclusion for sufferers, but patient and community level ownership of the program is essential for sustainability. More needs to be done to ensure that knowledge held at the health center is transferred to the patients and caregivers and repeated for new stakeholders. Research on the lived experience of affected persons, on the effectiveness of individual components of self-care, and on factors affecting patient adherence are urgently needed to guide service delivery. People faced with lifelong chronic disease deserve to be guided by appropriate and acceptable recommendations for self-care and provided with the necessary tools and support to achieve it.

The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.

The studies involving human participants were reviewed and approved by Liverpool School of Tropical Medicine Research Ethics Committee (approval no. 012-18), the Bangladesh Medical Research Council, and the Amhara Public Health Institute Research Ethics Review Committee. The participants provided their written informed consent to participate in this study.

Conceptualization, study design, methodology, manuscript preparation, JD, SM, HM, HB, LK-H, ASM, and FH. Contribution of resources, STJ, MJK, SMC, MT, MJT. Reviewing and editing MMR, AAK, AFR, AK, FS, TB, MN, KM, and GA. All authors contributed to the article and approved the submitted version.

This work was supported by funding from the Department for International Development (DFID) and GlaxoSmithKline (GSK) to the Centre for Neglected Tropical Diseases, Liverpool School of Tropical Medicine, for programmatic support for the elimination of lymphatic filariasis as a public health problem. Neither funder was involved in the study design, collection, analysis, interpretation of data, the writing of this article or the decision to submit it for publication.

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

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