Background
Neglected tropical diseases (NTDs) that result in lower-limb lymphoedema (i.e. swelling of the lower leg) include lymphatic filariasis (LF), leprosy and podoconiosis. Global burden estimates for people affected by LF lymphoedema, podoconiosis and leprosy are 15 million, 4 million and 2 to 3 million respectively [
1,
2]. Nationwide mapping in 2013 demonstrated that podoconiosis accounts for approximately 64.8%, LF for 13.2%, and leprosy for 12.8% of the total burden of lymphoedema in Ethiopia [
3].
Stigma related to lower-limb lymphoedema is one of the key issues that significantly increases its disease burden [
4] and acts as a major barrier to accessing morbidity management and disability prevention (MMDP) services [
5,
6]. The prejudice, discrimination and internalized stigma related to lower-limb lymphoedema not only compromise the psychological and social wellbeing of affected persons and their families but can also limit their access to healthcare and adherence to treatment [
5]. This can lead to a vicious cycle, creating further disability, illness, and reduced economic productivity [
7]. The three major sources of stigma for lower-limb lymphoedema are: i) misinformation about its causes, treatment and prevention amongst the community, affected persons and their families; ii) the associated poverty and reduced quality of life due to affected individuals’ lost economic productivity; and iii) the economic burden related to the costs of care, including transport to health facilities [
8‐
11]. People affected by podoconiosis, leprosy and LF are stigmatized in all areas of their daily life. Social functions such as partner selection and marriage, employment, and participation in community leadership are common domains of life in which affected persons and their families experience discrimination. These deep-seated stigmatizing experiences can cause internalized stigma in affected persons, i.e. acceptance of stigmatizing social views and resultant feelings of shame, guilt or fear, as a result of the experience of discrimination. Internalized stigma is manifested in the form of low self-esteem, suicidal ideation, and avoidance of interactions with non-affected community members. [
12]. Breaking the vicious cycle of stigma related to lymphoedema has therefore become a top priority for various community organizations and researchers. Strategies to address this stigma have included: i) educational interventions; ii) community-based socio-economic rehabilitation; and iii) providing integrated services in nearby health facilities at low or no cost [
13]. Interventions often aim to remove the drivers of stigma and address the norms and policies that facilitate the stigmatization process. The drivers of stigma include fear of infection through contact, concerns about productivity due to poor health, social judgment and blame. Facilitators of stigma include social isolation and lack of social support [
8,
14,
15].
Studies have suggested that multi-component interventions are more effective than single-component interventions for stigma reduction [
16,
17]. Multi-component stigma reduction interventions target both the stigmatizers and the stigmatized to ensure holistic and sustainable changes in outcomes. These interventions support individuals with health-related stigma to cope with experienced stigma (i.e. actual experiences of discrimination) and overcome internalized stigma, as well as reaching out to community members to shift harmful notions about health conditions through community dialogues or engaging local leaders to share anti-stigma messages [
18‐
21].
Implementing and donor actors now widely recognize that joint approaches to reduce stigma across NTDs may be feasible given the similarities in causes, manifestations and interventions [
22], but there remains a knowledge gap in regard to relevant, evidence-based stigma reduction interventions for use within integrated MMDP programmes. To address this knowledge gap, an implementation research project titled ‘Improving access to integrated Morbidity management and disability PREvention Services through Stigma reduction for people with lower limb lymphoedema in Ethiopia’ (IMPRESS) was implemented in selected districts of Northern Ethiopia where LF, podoconiosis and leprosy-related lymphoedema are prevalent. The IMPRESS project was embedded within the ‘Excellence in Disability Prevention Integrated across NTDs’ (EnDPoINT) programme, which ran from 2017 to 2021 in response to a request by the Federal Ministry of Health (FMOH) in Ethiopia. Details in regard to the development and implementation of the EnDPoINT/IMPRESS project have been provided in previous reports [
23]. The EnDPoINT care package entailed comprehensive MMDP and mental health services including capacity-building training for health care organization staff, case detection and treatment for patients at health facility level, integrated mental health care, and community awareness campaigns [
23‐
26]. The IMPRESS study built on this unique opportunity and focused on the specific question of how best to reduce stigma within this integrated MMDP programme. EnDPoINT/IMPRESS was guided by the World Health Organization’s Community-Based Rehabilitation (CBR) strategy [
27], as it promotes inclusion and participation of marginalized groups through multi-sectoral interventions across five key domains (health, education, livelihood, social, and empowerment). The CBR model acknowledges that programmes need to go beyond the health domain to empower affected persons to take an active role in their development, as it supports equity in services by building capacity amongst affected persons and their communities [
28]. The IMPRESS study embedded Community Conversation (CC) as one of several intervention components within EnDPoINT’s holistic care package [
23] with the aim to increase disease-related health literacy at the community level, reduce stigma and improve access to MMDP services. In other disease contexts including HIV and mental health, studies have documented the positive role of CC in stigma reduction [
29]. In our previous qualitative process evaluation report from IMPRESS [
30], the acceptability and feasibility of integrating CC into the lymphoedema care package and routine primary health care services was documented. This report also highlighted the potential role of CC in raising awareness and overcoming stigmatizing attitudes within the community. However, the role of CC in reducing stigma when added to the holistic MMDP care package had not been explored quantitatively. The study presented here therefore aimed to quantitatively investigate whether CC contributed significantly to stigma reduction when added into EnDPoINT’s integrated holistic care package.
Discussion
The stigma associated with the three skin NTDs studied is known to affect the psychosocial wellbeing of affected persons and their associates and obstructs access to health care services. Empirical evidence from other health conditions supports the importance of CC in reducing stigma through enabling participants to set a plan of action, develop a sense of common purpose, overcome fear, denial and passivity and move from being passive recipients of health information to active problem solvers [
29]. CC has been shown to be vital for enabling healthy behaviours, facilitating timely and appropriate access to health services and supporting optimum treatment adherence [
39,
40]. Some community-based organizations (CBOs) in Ethiopia have applied a CC approach to tackle stigma and promote access to lymphoedema care services [
41,
42], though quality of the implementation process and outcomes of these efforts have not been adequately documented. Though community-based organizations have also been implementing CC as a vertical program, little is known about the feasibility of integrating CC into the primary health care system in combination with MMDP services. Embedded within the EnDPoINT programme [
24], the IMPRESS project – to our knowledge – made the first effort of its kind to integrate CC into the primary health care system to tackle stigma related to lower-limb lymphoedema.
This study revealed findings that are relevant for decision makers, implementers and researchers. The intervention effect of CC was mixed. On the one hand, no difference was observed between the intervention and control districts in terms of improvements in knowledge about the causes of lymphoedema, stigmatizing attitudes and stigmatizing practice (perceived social distance and social support). These are the key drivers of social stigma in lymphoedema patients [
8,
9]. On the other hand, the time effects were significant in both the intervention and control districts. Changes were observed over a six-month period of CC implementation: community members’ knowledge about the cause(s) of lymphoedema improved, and stigmatizing attitudes and perceived social distance reduced. These changes may be accounted for directly or indirectly by the EnDPoINT holistic care package. The EnDPoINT pilot study (without CC) suggested an association between the holistic care package and improved quality of life, reduced levels of depression, and reduced experiences of internalized stigma and discrimination [
26]. A range of studies have also demonstrated that encouraging simple self-care measures to promote foot hygiene can reduce limb swelling and improve quality of life [
43‐
45].
Positive changes (reductions in stigmatizing attitudes, social distance, experienced and internalized stigma) were observed in intervention and control districts over time. Unexpectedly, these changes were larger in the control districts. This might be explained by low quality of implementation of CC in the intervention district. Our previous qualitative process evaluation [
30] documented a range of challenges affecting quality of CC implementation. Some of these challenges were: inadequacy of training and supportive supervision, perceived complexity of the CC facilitation guide, lack of competence to run CC sessions according to the principles and procedures in the guide, and low motivation and commitment of CC facilitators to run the CC sessions as planned due to absence of incentives. These challenges are likely to have compromised the quality of outcomes of the CC intervention, resulting in misunderstanding of health messages about lymphoedema. A study that applied a Lay Health Advisor model in southern Ethiopia also observed misunderstanding of health messages due to the low competence of Lay Health Advisors in communicating complex health messages to recipients [
46]. The smaller reduction in internalized and experienced stigma in CC intervention districts may also be associated with low competence of CC facilitators.
As this study reports changes observed over six-month follow-up, longer-term follow-up assessments may better capture the association between the implementation of the CC intervention and its outcomes. Hence, for broader understanding of the role of an integrated CC intervention as a stigma reduction strategy in the context of skin-NTDs, future research may consider longer-term follow-ups to assess the sustained impact of the quality of implementation of the CC intervention on stigma and community attitudes towards lower-limb lymphoedema. Additionally, though patients and community members participated actively in the design and implementation of the interventions, inadequate feedback mechanisms for ensuring their satisfaction with the process and outcomes of their engagement may be a limitation of this study.
An economic evaluation of the EnDPoINT care package is currently underway. This suggests that effectiveness of the EnDPoINT care package combined with the CC intervention may be slightly higher than usual care (i.e. the EnDPoINT care package alone), but that CC is unlikely to be good value for money given the high values of incremental cost-effectiveness ratio (ICER), i.e. cost per one unit change in effectiveness outcome (N. Ivashikina, Personal Communication). Coupled with the implementation challenges reported previously [
30], the high economic cost of integrating CC into the primary health care system is likely to be an additional burden in resource-constrained settings.
As this study addressed only the added value of CC to the EnDPoINT care package in reducing stigma related to lower limb lymphoedema, our findings should be interpreted only in the context of MMDP services integrated into primary health facilities for lymphoedema patients, rather than as standalone intervention. The unexpected observations, particularly in control districts, do not nullify the documented positive role of standalone CC interventions implemented in community settings, for example in competence levels in HIV [
29,
39,
40].
In conclusion, adding CC to a holistic care package integrated into the primary health care system may not result in any further gains in tackling stigma related to lymphoedema than those already brought about by the care package. Hence, policy-makers and practitioners may prioritize integration of the EnDPoINT care package into the primary health care system instead of the CC intervention for efficient utilization of financial and human resources.
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