Meaningful Engagement of Persons Affected by Leprosy in Research: An Exploration of Its Interpretation, Barriers, and Opportunities.

Despite the growing interest in public and patient involvement in research, best practices in the leprosy context have yet to be explored. This mixed-method study aimed to explore the interpretation, barriers and opportunities of meaningful engagement of persons affected by leprosy in research through: (i) an exploratory phase consisting of key informant interviews with experts in public and patient involvement (n = 2) and experts-by-experience (i.e., persons affected by leprosy; n = 4), and (ii) an in-depth phase among leprosy researchers consisting of an online survey (n = 21) and key informant interviews (n = 7). Qualitative data were thematically analyzed. Basic descriptive statistics were used to summarize the survey data. Key informant interviewees unanimously agreed to the importance of engagement in research. Survey results indicated that the level of engagement differed across research stages. Identified barriers included a lack of skills for or awareness of engagement among both experts-by-experience and researchers, stigma and limited time and resources. Opportunities included capacity strengthening, creating a shared understanding, building rapport, and establishing a safe environment. In conclusion, this exploratory study emphasized the importance of engagement of experts-by-experience in leprosy research and identified ways forward that include, but are not limited to, the acknowledgement of its value and creating a shared understanding.

Experiences of living with leprosy: A systematic review and qualitative evidence synthesis.

OBJECTIVE: The objective of the review was to identify, appraise, and synthesise qualitative studies on the lived experience of individuals diagnosed with leprosy, the impact of the disease, and how they coped with the disease burden. INTRODUCTION: Leprosy is a chronic disease with long-term biopsychosocial impact and is a leading cause of preventable disabilities. It traps the individuals with leprosy in a vicious circle of disease, stigma, and poverty. The efforts to reduce stigma and discrimination and improve their quality of life have not kept pace with the success of the multidrug treatment. INCLUSION CRITERIA: This review considered published literature on the lived experience of individuals diagnosed with leprosy. There were no limitations on gender, background, or country. All qualitative or mixed-methods studies were accepted. METHODS: The review followed the JBI meta-aggregation approach for qualitative systematic reviews. A structured literature search was undertaken using multiple electronic databases: PubMed, Embase, Web of Science, and CINAHL. RESULTS: The search identified 723 publications, and there were 446 articles after deduplication. Forty-nine studies met the inclusion criteria. The final 173 findings were synthesised into ten categories and aggregated into four synthesised findings: biophysical impact, social impact, economic impact, and mental and emotional impact. These synthesised findings were consistent across the included studies from a patient's perspective. The way people coped with leprosy depended on their interpretation of the disease and its treatment. It affected their help-seeking behaviour and their adherence to treatment and self-care. The review has identified a multi-domain effect on the affected individuals, which goes beyond the biological and physical effects, looking at the social issues, specific difficulties, emotions, and economic hardships. CONCLUSIONS: The researchers, health professionals, and policymakers could use the synthesised findings to address the concerns and needs of the leprosy-affected individuals and offer appropriate support to manage their lives. SYSTEMATIC REVIEW REGISTRATION NUMBER: PROSPERO Registration number: CRD42021243223.

Understanding leprosy reactions and the impact on the lives of people affected: An exploration in two leprosy endemic countries.

BACKGROUND: Leprosy reactions, Type-1 and erythema nodosum leprosum, are immune-mediated complications of leprosy, which play a significant role in the morbidity associated with the disease. A considerable amount of literature has been published on the impact of leprosy in general but few studies focus specifically on leprosy reactions. This study aimed to investigate the impact of leprosy reactions on physical, psychological, and social aspects of the lives of people affected by analysing their life experiences and perspectives about leprosy reactions. METHODS/PRINCIPAL FINDINGS: This qualitative study involved people affected by leprosy reactions and their family members in two leprosy endemic countries. The data were collected through 66 interviews and 9 focus group discussions (4-6 participants each) in Surabaya, Indonesia, and Purulia, India. Content analysis and conversational analysis were performed. This study found that both types of leprosy reactions were perceived as an unpredictable and painful condition. Leprosy reactions restricted physical activities of the participants, such as going to bathroom, sleeping, eating, and cooking. In the interviews, the respondents expressed a range of emotions and feelings including confusion, sadness, anxiety, and anger. Some recounted that they felt stigmatized and lost opportunities to socialise and earn money. Differences between the two settings were identified. The majority of Indonesian participants preferred to stay at home, and some concealed the diagnosis of leprosy, while most of the Indian respondents continued working up to the time of hospitalization. CONCLUSION: Leprosy reactions are a distressing complication of leprosy and adversely affect the lives of those affected. Individuals reported physical discomfort, distress, anxiety, stigma, and financial hardship and these negative impacts in the physical, psychological, and social spheres reinforced each other. These findings provide important information about a need for early detection and sustained commitment to follow-up care for people with a history of leprosy reactions. More research on new drugs for reactional episodes, tools to measure knowledge, attitude, and practice, and costing study on leprosy reactions treatment are needed. We recommend the development and testing of holistic strategies to improve the management of leprosy reactions.

How do Positive Deviants Overcome Health-Related Stigma? An Exploration of Development of Positive Deviance Among People With Stigmatized Health Conditions in Indonesia.

A constructivist grounded theory approach was used to understand how some people living with stigmatized health conditions develop positive deviance to overcome stigma. We examined interviews from 13 identified positive deviants living with four different stigmatized health conditions (HIV, leprosy, schizophrenia, and diabetes) in Indonesia. Positive deviance develops in the form of psychological empowerment through improvement of self-belief and perception (intrapersonal component), development of understanding and skill to exert control in life (interactional component), and self-discovery of successful behaviors and strategies to avert stigma (behavioral component). Positive deviants, after being empowered, start empowering others affected by sharing their knowledge and fostering social awareness and acceptance. The findings revealed the presence of problem-solving ability and agency within the community of stigmatized individuals in Indonesia and warrant researchers to partner with the community to expedite the diffusion of transferable positive deviant strategies within and outside the communities.

Assessing the prospect of a common health-related stigma reduction response: Cross-perspectives of people living with stigmatised health conditions in Indonesia.

ABSTRACTThis study explored the possibility of a common health-related stigma reduction intervention among people living with HIV, leprosy, schizophrenia and diabetes in Indonesia by assessing their perspectives towards others with the same (within group) and different health conditions (across groups), and willingness to participate in such a program. This mixed-methods study was conducted in West Java, Indonesia between March and June 2018. Eighty participants completed a survey with social distance scale (SDS), while 12 focus group discussion were conducted. Participants with HIV, leprosy and diabetes reported lower within-group SDS scores (4.14 ± 3.65; 4.25 ± 3.95; 7.23 ± 5.31, respectively) while those with schizophrenia reported the highest within-group SDS score (7.76 ± 4.63). Participants with diabetes reported a twofold higher across-group SDS score towards people with the other three health conditions (p < 0.05). The qualitative findings showed that the perception of participants towards one another was shaped by knowledge, understanding and relatedness to the experience of living with health-related stigma. Overall, participants supported the idea of a common stigma reduction intervention for different health conditions, but recommended step-wise implementation of such interventions. Accordingly, this study recommends piloting a common stigma reduction intervention with special focus on fostering understanding, awareness and empathy between people living with different health conditions.

Intersectionality and health-related stigma: insights from experiences of people living with stigmatized health conditions in Indonesia.

BACKGROUND: Health-related stigma is a complex phenomenon, the experience of which intersects with those of other adversities arising from a diversity of social inequalities and oppressive identities like gender, sexuality, and poverty - a concept called "intersectionality". Understanding this intersectionality between health-related stigma and other forms of social marginalization can provide a fuller and more comprehensive picture of stigma associated with health conditions. The main objective of this paper is to build upon the concept of intersectionality in health-related stigma by exploring the convergence of experiences of stigma and other adversities across the intersections of health and other forms of social oppressions among people living with stigmatized health conditions in Indonesia. METHODS: This qualitative study interviewed 40 people affected by either of four stigmatizing health conditions (HIV, leprosy, schizophrenia, and diabetes) in Jakarta and West Java, Indonesia between March and June 2018. Data was analyzed thematically using an integrative inductive-deductive framework approach. RESULTS: The main intersectional inequalities identified by the participants were gender and socioeconomic status (n = 21), followed by religion (n = 13), age (n = 11), co-morbidity (n = 9), disability (n = 6), and sexuality (n = 4). Based on these inequalities/identities, the participants reported of experiencing oppression because of prevailing social norms, systems, and policies (macro-level), exclusion and discrimination from societal actors (meso-level), and self-shame and stigma (micro-level). While religion and age posed adversities that negatively affected participants in macro and meso levels, they helped mitigate the negative experiences of stigma in micro level by improving self-acceptance and self-confidence. CONCLUSION: This study uncovered how the experience of health-related stigma intersects with other oppressions originating from the various social inequalities in an individual's life. The findings highlight the importance of acknowledging and understanding the multi-dimensional aspect of lives of people living with stigmatized health conditions, and warrant integrated multi-level and cross-cutting stigma reduction interventions to address the intersectional oppressions they experience.

Validation of a cross-NTD toolkit for assessment of NTD-related morbidity and disability. A cross-cultural qualitative validation of study instruments in Colombia.

BACKGROUND: Many neglected tropical diseases (NTDs) are not fatal, but they are disabling, disfiguring and stigmatizing. More accurate data on these aspects would benefit planning, monitoring and evaluation of interventions, as well as provision of appropriate services for the often life-long consequences. In 2015, a cross-NTD toolkit was developed, consisting of a variety of existing questionnaires to measure morbidity, disability and health-related quality of life. The toolkit covers the domains of the International Classification of Functioning, Disability and Health (ICF) framework. These tools have been developed in a source country, however, it was intended for the cross-NTD toolkit to be applicable across NTDs in many countries with different cultures and languages in order to generate universally comparative data. Therefore; the present study aimed to validate several tools of the toolkit among people affected by leprosy or leishmaniasis in the cultural settings of Cartagena and Cúcuta, Colombia. METHODOLOGY: This study aimed to validate the following tools among 55 participants between 18-85 years old, affected by leprosy and leishmaniasis: (I) Clinical Profile, (II) Self-Reporting Questionnaire (SRQ), (III) WHO Quality of Life assessment-abbreviated version (WHOQOL-BREF), and (IV) WHO Quality of Life assessment-Disability (WHOQOL-DIS). The tools were administered during face-to-face interviews and were followed by open questions about the respondents' thoughts on format of the tool and the understanding, relevance and acceptability of the items. The tools were validated using a qualitative method approach based on the framework for cultural equivalence, measured by the cultural, item, semantic and operational equivalences. RESULTS: The Clinical Profile was seen as acceptable and relevant, only the semantic equivalence was not as satisfying and needs a few adaptations. The SRQ was very well understood and shows to reach the equivalences for the population of Colombia without any additional changes. Several items of the WHOQOL-BREF and the WHOQOL-DIS were not well understood and changes are recommended due to semantic difficulties. Operational equivalence of both questionnaires was not as desired in relation to the used response scales. The participants shared that the tools are relevant and important for their particular situation. CONCLUSIONS/SIGNIFICANCE: The SRQ is found to be a valid tool for Colombia and can be included in the cross-NTD toolkit. The Clinical Profile, WHOQOL-BREF & WHOQOL-DIS need changes and retesting among Colombian people affected by an NTD. The toolkit as a whole is seen as useful to show the effects leprosy and leishmaniasis have on the participants. This cultural validation will contribute to a universally applicable cross-NTD toolkit.

Assessing the Impact of the Twin Track Socio-Economic Intervention on Reducing Leprosy-Related Stigma in Cirebon District, Indonesia.

The consequences of leprosy go beyond the physical, social and psychological, as leprosy can drive persons affected and their families into poverty, stigmatization and disability. This paper describes the impact of a socio-economic development (SED) intervention that uses a twin-track approach (two micro-credit models) to reduce leprosy-related stigma in Cirebon District, Indonesia. A randomized-controlled mixed-methods study design was used to test the effectiveness of the SED intervention. Three scales were used to measure stigma and participation restrictions among 30 SED clients and 57 controls, 20 in-depth interviews with SED clients and seven Focus Group Discussions (FGDs) with key persons were held and 65 profiles of the clients were written up and analysed. The qualitative data shows the socio-economic status of 44 out of 65 SED clients (67%) improved. The median family income increased by 25%, more clients reported higher self-esteem, better interaction with neighbours and less stigma than before, although disclosure concerns remained an issue. The scales indicate a positive effect of the intervention on reducing stigma (e.g., Stigma Assessment and Reduction of Impact (SARI) stigma scale mean difference total score of pre and post assessment for SED clients versus the control group was 8.5 versus 5.6). A twin track socio-economic intervention, if embedded and integrated, can increase participation, and be constructive in reducing leprosy-related stigma.

Impact of socio-economic development, contact and peer counselling on stigma against persons affected by leprosy in Cirebon, Indonesia ­a randomised controlled trial.

Objectives: People affected by leprosy are often stigmatised, but stigma is rarely quantified and the effectiveness of interventions is often not evaluated. The SARI Project aimed to test and evaluate three interventions: counselling (involving peer counsellors), socio-economic development (SED) and contact between community members and affected people. Results: This study used a controlled trial design in which pairs of the stigma-reduction interventions were randomly allocated to sub-districts in Cirebon District,Indonesia. The study sample consisted of one cohort of people affected by leprosy (on treatment or treated) and two independent samples of community members. The latter were selected through purposive sampling. Three scales (e.g. SARI Stigma Scale, Participation scale) were applied among leprosy-affected people and two scales (e.g. Social Distance Scale) were used among community members pre- and post-intervention. Among affected people (n=237), significant differences in reduction of stigma and participation restrictions were found in all intervention areas and an improvement in quality of life in some intervention areas. Social distance and social stigma significantly reduced among community members (n=213 and 375) in the two intervention areas where the contact intervention was implemented. Two of the five instruments indicated changes in the control area, but the changes in the intervention areas were much larger. Conclusion: The SARI Project has demonstrated that a measurable reduction in leprosy-related stigma can be achieved, both at community level and among people affected by leprosy, using reproducible interventions that can be adapted to different settings and target groups.

Cultural validation of a new instrument to measure leprosy-related stigma: the SARI Stigma Scale.

Background: There is a need for comprehensive, valid and reliable instruments to assess leprosy-related stigma. This paper presents the process of the cross-cultural validation of an instrument in Cirebon District, Indonesia initiated by the Stigma Assessment and Reduction of Impact (SARI) project. Methods: The Berger Scale was initially developed to assess HIV/AIDS-related stigma. This study explores the conceptual, item, semantic, operational and measurement equivalence of this scale for leprosy. The process included a qualitative study, translation and back-translation, training of interviewers, a pilot and the main data collection. We aimed for a sample of 154 people affected by leprosy with 60 repeat interviews. They were selected through convenience sampling. Results: The original scale showed acceptable conceptual equivalence, but insufficient item, semantic and operational equivalences. For instance, there were irrelevant HIV-related items and the respondents found it difficult to indicate their level of agreement with the given statements. Major adjustments were necessary, leading to a new version of the scale. The measurement properties of the new version showed good internal consistency (Cronbach's alpha 0·88); no floor or ceiling effects; and a good reliability (intra-class correlation coefficient 0·75).

A Cluster-Randomized Controlled Intervention Study to Assess the Effect of a Contact Intervention in Reducing Leprosy-Related Stigma in Indonesia.

BACKGROUND: Can deliberate interaction between the public and persons affected by leprosy reduce stigmatization? The study described in this paper hypothesises that it can and assesses the effectiveness of a 'contact intervention'. METHODS/PRINCIPAL FINDINGS: This cluster-randomized controlled intervention study is part of the Stigma Assessment and Reduction of Impact (SARI) project conducted in Cirebon District, Indonesia. Testimonies, participatory videos and comics given or made by people affected by leprosy were used as methods to facilitate a dialogue during so-called 'contact events'. A mix of seven quantitative and qualitative methods, including two scales to assess aspects of stigma named the SDS and EMIC-CSS, were used to establish a baseline regarding stigma and knowledge of leprosy, monitor the implementation and assess the impact of the contact events. The study sample were community members selected using different sampling methods. The baseline shows a lack of knowledge about leprosy, a high level of stigma and contrasting examples of support. In total, 91 contact events were organised in 62 villages, directly reaching 4,443 community members (mean 49 per event). The interview data showed that knowledge about leprosy increased and that negative attitudes reduced. The adjusted mean total score of the EMIC-CSS reduced by 4.95 points among respondents who had attended a contact event (n = 58; p < 0.001, effect size = 0.75) compared to the score at baseline (n = 213); for the SDS this was 3.56 (p < 0.001, effect size = 0.81). About 75% of those attending a contact event said they shared the information with others (median 10 persons). CONCLUSIONS/SIGNIFICANCE: The contact intervention was effective in increasing knowledge and improving public attitudes regarding leprosy. It is relatively easy to replicate elsewhere and does not require expensive technology. More research is needed to improve scalability. The effectiveness of a contact intervention to reduce stigma against other neglected tropical diseases and conditions should be evaluated.

Lay and peer counsellors to reduce leprosy-related stigma--lessons learnt in Cirebon, Indonesia.

OBJECTIVE: Counselling has been identified as a promising strategy to reduce stigma. Lay and peer counsellors have provided counselling in various fields, but this has not yet been studied in the field of leprosy. The Stigma Assessment and Reduction of Impact (SARI) project in Cirebon District, Indonesia took up this endeavour. This paper describes the initial experiences based on the perspectives of the lay and peer counsellors and aims to provide lessons learnt for future initiatives. METHODS: The selection of lay and peer counsellors was based upon pre-defined criteria such as completed junior high school and level of confidence. This study draws on the notes of seven monitoring and evaluation meetings and 21 group discussions the main researcher facilitated with the lay and peer counsellors and the notes written by the lay and peer counsellors on the sessions with their clients. RESULTS: In total, 198 people affected by leprosy were offered counselling by the 11 lay and 12 peer counsellors; 145 accepted this offer. The other 53 either did not need counselling or did not want to participate for example due to worries about disclosure. Effective communication skills such as listening and asking effective questions were important, but also difficult to acquire for the lay and peer counsellors. Sharing personal experiences was highly appreciated by clients and stimulated a deepened reflection. CONCLUSION: Challenges related to concealment and effective skills exist, but some people affected by leprosy and others can become effective counsellors making it at the outset a challenging but nevertheless promising intervention.

Dealing with stigma: experiences of persons affected by disabilities and leprosy.

Persons affected by leprosy or by disabilities face forms of stigma that have an impact on their lives. This study seeks to establish whether their experiences of stigma are similar, with a view to enabling the two groups of people to learn from each other. Accounts of experiences of the impact of stigma were obtained using in-depth interviews and focus group discussion with people affected by leprosy and by disabilities not related to leprosy. The analysis shows that there are a lot of similarities in impact of stigma in terms of emotions, thoughts, behaviour, and relationships between the two groups. The main difference is that those affected by leprosy tended to frame their situation in medical terms, while those living with disabilities described their situation from a more social perspective. In conclusion, the similarities offer opportunities for interventions and the positive attitudes and behaviours can be modelled in the sense that both groups can learn and benefit. Research that tackles different aspects of stigmatization faced by both groups could lead to inclusive initiatives that help individuals to come to terms with the stigma and to advocate against exclusion and discrimination.

The cultural validation of two scales to assess social stigma in leprosy.

BACKGROUND: Stigma plays in an important role in the lives of persons affected by neglected tropical diseases, and assessment of stigma is important to document this. The aim of this study is to test the cross-cultural validity of the Community Stigma Scale (EMIC-CSS) and the Social Distance Scale (SDS) in the field of leprosy in Cirebon District, Indonesia. METHODOLOGY/PRINCIPLE FINDINGS: Cultural equivalence was tested by assessing the conceptual, item, semantic, operational and measurement equivalence of these instruments. A qualitative exploratory study was conducted to increase our understanding of the concept of stigma in Cirebon District. A process of translation, discussions, trainings and a pilot study followed. A sample of 259 community members was selected through convenience sampling and 67 repeated measures were obtained to assess the psychometric measurement properties. The aspects and items in the SDS and EMIC-CSS seem equally relevant and important in the target culture. The response scales were adapted to ensure that meaning is transferred accurately and no changes to the scale format (e.g. lay out, statements or questions) of both scales were made. A positive correlation was found between the EMIC-CSS and the SDS total scores (r=0.41). Cronbach's alphas of 0.83 and 0.87 were found for the EMIC-CSS and SDS. The exploratory factor analysis indicated for both scales an adequate fit as unidimensional scale. A standard error of measurement of 2.38 was found in the EMIC-CSS and of 1.78 in the SDS. The test-retest reliability coefficient was respectively, 0.84 and 0.75. No floor or ceiling effects were found. CONCLUSIONS/SIGNIFICANCE: According to current international standards, our findings indicate that the EMIC-CSS and the SDS have adequate cultural validity to assess social stigma in leprosy in the Bahasa Indonesia-speaking population of Cirebon District. We believe the scales can be further improved, for instance, by adding, changing and rephrasing certain items. Finally, we provide suggestions for use with other neglected tropical diseases.

The meaning of leprosy and everyday experiences: an exploration in cirebon, indonesia.

It is imperative to consider the meaning of leprosy and everyday experiences of people affected by leprosy and key persons in the community if one aims to make leprosy services more effective, which appears necessary in Indonesia given the large numbers of new cases detected annually. However, little is written in the international literature about the experiences of people currently being treated for leprosy, those cured, or other key informants. This paper analyses the narratives of the people by drawing upon in-depth interviews with 53 participants and 20 focus groups discussions. The participants were purposively selected. We provide insights into the experiences of people and the meaning they give to leprosy and highlight aspect of aetiology, spirituality, religion, darkening of the skin, and sorcery. We also examine experiences of seeking care and focused on the impact of the disease in particular on the elderly and children. In conclusion, the continued need for implementation of leprosy services in Indonesia is very evident. The diversities in people's experiences with leprosy indicate a demand for responsive leprosy services to serve the diverse needs, including services for those formally declared to be "cured."