Macintyre, K; Bakker, MI; Bergson, S; Bhavaraju, R; Bond, V; Chikovore, J; Colvin, C; Craig, GM; Cremers, AL; Daftary, A; Engel, N; France, NF; Jaramillo, E; Kimerling, M; Kipp, A; Krishnaratne, S; Mergenthaler, C; Ngicho, M; Redwood, L; Rood, EJJ; Sommerland, N; Stangl, A; van Rie, A; van Brakel, W; Wouters, E; Zwerling, A; Mitchell, EMH (2017) Defining the research agenda to measure and reduce tuberculosis stigmas. The international journal of tuberculosis and lung disease, 21 (11). pp. 87-96. ISSN 1027-3719 DOI: https://doi.org/10.5588/ijtld.17.0151 Downloaded from: http://researchonline.lshtm.ac.uk/4558189/ DOI: 10.5588/ijtld.17.0151 Usage Guidelines Please refer to usage guidelines at http://researchonline.lshtm.ac.uk/policies.html or alternatively contact researchonline@lshtm.ac.uk. Available under license: http://creativecommons.org/licenses/by/2.5/ INT J TUBERC LUNG DIS 21(11):S87–S96 Q 2017 The Union http://dx.doi.org/10.5588/ijtld.17.0151 SUPPLEMENT: TB STIGMA Defining the research agenda to measure and reduce tuberculosis stigmas K. Macintyre,* M. I. Bakker,† S. Bergson,‡ R. Bhavaraju,§ V. Bond,¶# J. Chikovore,** C. Colvin,†† G. M. Craig,‡‡ A. L. Cremers,§§ A. Daftary,¶¶ N. Engel,## N. Ferris France,*** E. Jaramillo,††† M. Kimerling,‡ A. Kipp,‡‡‡ S. Krishnaratne,¶ C. Mergenthaler,† M. Ngicho,‡ L. Redwood,§§§ E. J. J. Rood,† N. Sommerland,¶¶¶ A. Stangl,### A. van Rie,**** W. van Brakel,†††† E. Wouters,### A. Zwerling,‡‡‡‡ E. M. H. Mitchell‡ *Oxford Health Evaluations, Oxford, UK; †KIT Royal Tropical Institute, Amsterdam; ‡KNCV Tuberculosis Foundation, The Hague, The Netherlands; §Global Tuberculosis Institute, Rutgers, The State University of New Jersey, Newark, New Jersey, USA; ¶London School of Tropical Medicine & Hygiene, London, UK; #Zambart, University of Zambia, Lusaka, Zambia; **Human Sciences Research Council, Durban, South Africa; ††US Agency for International Development Global Health Bureau, Washington DC, USA; ‡‡Centre for Research in Primary and Community Care, School of Health and Social Work, University of Hertfordshire, Hatfield, UK; §§Amsterdam Institute for Global Health and Development, Amsterdam, The Netherlands; ¶¶Department of Epidemiology, McGill University, Montreal, Quebec, Canada; ##Department of Health, Ethics & Society/Care And Public Health Research Institute, University of Maastricht, Maastricht, The Netherlands; ***The Work for Change/Irish Forum for Global Health, Dublin, Ireland; †††Global TB Programme, World Health Organization, Geneva, Switzerland; ‡‡‡ Institute for Medicine and Public Health, Vanderbilt University, Nashville, Tennessee, USA; §§§University of Sydney, Sydney, New South Wales, Australia; ¶¶¶Department of Sociology, Centre for Longitudinal and Life Course Studies, University of Antwerp, Antwerp, Belgium; ###International Center for Research on Women, Stigma Action Network Secretariat, Washington DC, USA; ****University of Antwerp, Antwerp, Belgium; †††† Netherlands Leprosy Relief, Amsterdam, The Netherlands; ‡‡‡‡Department of Epidemiology, University of Ottawa, Ontario, Canada SUMMARY Crucial to finding and treating the 4 million tuberculosis (TB) patients currently missed by national TB programmes, TB stigma is receiving well-deserved and longdelayed attention at the global level. However, the ability to measure and evaluate the success of TB stigmareduction efforts is limited by the need for additional tools. At a 2016 TB stigma-measurement meeting held in The Hague, The Netherlands, stigma experts discussed and proposed a research agenda around four themes: 1) drivers: what are the main drivers and domains of TB stigma(s)?; 2) consequences: how consequential are TB stigmas and how are negative impacts most felt?; 3) burden: what is the global prevalence and distribution of TB stigma(s) and what explains any variation? 4): intervention: what can be done to reduce the extent and impact of TB stigma(s)? Each theme was further subdivided into research topics to be addressed to move the agenda forward. These include greater clarity on what causes TB stigmas to emerge and thrive, the difficulty of measuring the complexity of stigma, and the improbability of a universal stigma ‘cure’. Nevertheless, these challenges should not hinder investments in the measurement and reduction of TB stigma. We believe it is time to focus on how, and not whether, the global community should measure and reduce TB stigma. K E Y W O R D S : discrimination; human rights; social justice; respect; equity TUBERCULOSIS (TB) STIGMA is beginning to receive some well-deserved attention at the global level.1 ‘Zero suffering’ is one of the three goals of the World Health Organization’s End TB Strategy, and reference to stigma is common in key implementation documents.2 The Global Fund has declared TB stigma to be among the most commonly identified barriers to fighting the epidemic,3 and United Nations agencies have called for an end to discrimination in health care.1 Many experts believe that it will be difficult to find the estimated 4 million missing TB cases without addressing TB stigma.1,4 Experts also believe that the full potential of new drugs and regimens for multidrug-resistant TB (MDR-TB), paediatric TB and latent tuberculous infection (LTBI) cannot be realised without addressing TB stigma. Increasingly, countries using Global Fund support are attempting to reduce TB stigma and discrimination across and within elements of their TB programmes. However, Correspondence to: Ellen M H Mitchell, Evidence Team, KNCV Tuberculosis Foundation, Benoordenhoutseweg 46, The Hague 2596 BC, The Netherlands. e-mail: ellen.mitchell@kncvtbc.org Article submitted 28 February 2017. Final version accepted 6 August 2017. S88 The International Journal of Tuberculosis and Lung Disease the capacity to measure and evaluate the success of these interventions needs to be fostered.5–8 To begin to address this gap, an expert meeting took place in May 2016 to discuss the measurement issues surrounding TB stigma. This article outlines a research agenda for improving the measurement of and action on TB stigma, and is a product of that meeting. The ultimate aim of this research agenda is catalytic. We want researchers, TB activists, and programme managers to measure TB stigma appropriately and enable them to design, implement and evaluate interventions to reduce the impact TB stigma has on patients, families, health workers, communities and the epidemic itself. While there are many ways to define stigma, we draw on Weiss et al.’s definition: ‘a social process, experienced or anticipated, characterised by exclusion, rejection, blame or devaluation, that results from experience, perception, or reasonable anticipation of an adverse social judgment about a person or group’.9 METHOD Expert meeting At a 3-day TB stigma measurement meeting hosted by the KNCV Tuberculosis Foundation in The Hague, The Netherlands, in 2016, stigma experts gathered to discuss the status of TB stigma measurement science and define which steps might be taken to improve tracking of TB stigma dynamics. Using free mapping, small group discussions and plenary debate, the group distilled the priorities and the proposed study designs or methodologies for the main research priorities summarised under four themes: Theme 1 (drivers): what are the main drivers and domains of TB stigma(s)? Theme 2 (consequences): how consequential are TB stigmas and how are TB stigma(s) impacts felt? Theme 3 (burden): what is the global prevalence and distribution of TB stigma(s) and what explains any variation? Theme 4 (intervention): what can be done to reduce the extent and impact of TB stigma(s)? General principles In advance of the meeting, we leveraged information from studies already conducted on TB stigma, and commissioned four new studies, including two systematic reviews of published stigma scales and interventions.10–13 Link et al., Weiss et al., Pescosolido et al. and others have described the extent of how TB stigma can be observed, felt and resisted at different levels of society.9,14–19 TB stigma can also be present at the level of societal discourse where cultural and social determinants may be more or less important.14,20 We also learned from research on stigmas other than for TB, with the caveat that not everything will translate readily. Finally, we built on the work that has outlined the typologies of stigma,20,21 which are produced in different ways.9 ‘Anticipated stigma’ (perceived stigma) is the worry that one will be devalued or tainted if one is found to have a TB diagnosis. While these fears may not actually materialise, anticipated stigma may interfere with care seeking and treatment adherence. ‘Secondary stigma’ refers to the taint that care givers, friends, family members, health workers or those in TB-associated industries (e.g., miners), may experience because of their link to TB or TB patients.22,23 ‘Internalised stigma’ or ‘self-stigma’ captures the idea that individuals may come to endorse negative attitudes about themselves, and therefore behave or think according to these negative messages.24 ‘Enacted stigma’ refers to behaviours, including microaggression, discrimination, rejection, isolation and denial of rights. It is useful to distinguish between enacted stigma and experienced stigma as two sides of the same coin, seen from the perspective of either the stigmatiser (enacted) or the stigmatised (experienced). Finally, ‘structural stigma’ describes laws, policies and institutional architecture that may be prejudicial or denigrating. RESULTS We developed a matrix of types of stigma and the populations that may produce them or be affected by stigma. We separated types of stigma (anticipated, courtesy, internalised/self, enacted or experienced and structural) from the drivers and consequences of stigma. The drivers of stigma(s) are factors that facilitate the stigmatisation process (e.g., infectiousness of TB, power differentials, ignorance, discriminatory laws). The consequences of stigma (e.g., nondisclosure, poor adherence, depression, stress, poor health-seeking behaviour, self-isolation, reduced quality of life, social distancing, forced isolation) must also be captured and measured, and need to be separated from the drivers or causes of stigma. Based on this approach, we considered a matrix of types of TB stigma and the populations that may produce them or be affected by stigma. Table 1 links the different forms of stigma and the specific groups (dyads) involved in producing them. A more complete picture would be three-dimensional, including the social and political spaces in which TB stigma(s) are enacted (Table 1). Research agenda for the measurement of TB stigma For each of the four key research thematic areas, we defined the main research questions, foundation studies, levels or subpopulations and study designs or methodologies most fitting to address these questions. One conclusion reached was that much of the existing TB stigma research has focused on TB patients (individuals who made it into a health Defining the research agenda Table 1 S89 Matrix of illustrative scales used in referenced studies, by type of TB stigma and affected population General public Subgroups at higher risk of TB TB patients Health care workers Policy makers Anticipated stigma Courtesy stigma 25–27 28 13 Internalised/ self-stigma Enacted or experienced stigma (discrimination) 15,29–31 33 32 33 Structural stigma (policies, laws, architecture, human rights) STP legal environment assessment TB ¼ tuberculosis; STP ¼ STOP TB Partnership. system). There may thus be much we still do not know about the prevalence, degree and impact of TB stigma among missing cases: those who fail to make it into the facilities. We therefore propose that future work should cast a wider net to include a broader set of actors, including the general public, community leaders, media, policy makers, health care workers and community health workers (HCWs/CHWs), and explore protective as well as risk factors. Theme I: What are the main drivers and domains of TB stigma(s)? To address this theme, one must articulate the complex constructs underlying stigma. So far, indepth qualitative work around why and how stigma emerges and which facilitators sustain it remains scarce. As one reviewer notes, ‘most authors on TB stigma seem to point to the fundamental cause of the stigma being the perceived contagiousness of TB’.34,35 In contrast, in their study of TB stigma in five health systems in Nicaragua, Macq et al. found that the ‘determinants of stigma . . . were the content and channels of information, and issues of domination and power.’36 A persistent quandary around disentangling TB stigma from other stigmatised identities hinders study of the drivers of TB stigma. In other words, it remains unclear how and when the roots of TB stigma differ from other stigmatised identities such as having human immunodeficiency virus (HIV) infection, being poor, homeless, an immigrant, or being dependent on drugs or alcohol.37,38 Some argue that TB stigma is hard to distinguish from xenophobia, elitism and racism.9,39 Other researchers suggest it is so bound up with HIV stigma that trying to tease them apart may be a fool’s errand.13,40–42 Specific drivers may depend on the type of stigma, setting, population and type of TB. Structural stigmas can be driven by power differentials, whereas anticipated stigma thrives on misinformation. Drivers may also differ by epidemic characteristics (low- or high-burden settings) or by the populations impacted (age, sex and class).9,14,43,44 Stigma toward people with MDR-TB and extensively drug-resistant TB (XDR-TB) and those with LTBI is likely fostered and sustained by distinct sets of underlying factors. The drivers of MDR-TB stigma may be weighted distinct- ly from drug-susceptible TB stigma in three ways. First, one of the classical facets of any stigma construct is the social designation of ‘dangerousness’. If a person has MDR- or XDR-TB, doubts about curability may fuel the social construction of MDRTB patients as embodying a mortal risk to others. The second classical facet of stigma is the conscious attachment of blame for one’s mark or condition.19,45 In the case of MDR-TB, there may be treatmentadherence behaviours that may at times contribute to the development of drug resistance, creating rhetorical opportunities to blame individuals for their disease. Third, MDR-TB patients are more likely to experience perception-altering side effects as a consequence of their treatment and catastrophic costs due to the length of their treatment, both of which can heighten their vulnerability to stigma.46 Hearing loss, psychological side effects and impoverishment can reinforce the social construction of MDR-TB patients as ‘deviant’, ‘unpredictable’ and ‘other’. Given the complexity of TB stigma, we believe that qualitative research before, during or after quantitative research is essential to ensure that the nuance and meanings are not lost in the numbers (Table 2). Theme II: How consequential are TB stigmas? How are TB stigma(s) impacts felt? Understanding when and how TB stigma is problematic is vital. Figure 1 illustrates the potential impacts of stigma along a person’s trajectory of TB infection, disease and recovery. Assessing stigma at the individual behavioural level is crucial to determine how TB stigma hampers case finding and recovery from TB or MDR-TB. A recent systematic review of TB scales concluded that researchers should optimise existing TB stigma scales and develop new ones to better capture specific micro-, meso- and macro-level constructs (Figure 1).53 We recognise that TB stigma interacts on complex levels with psychological, social and political factors.41,43,54 The impact of stigma is therefore not unidirectional and not always negative. For example, stigma can make some patients more adherent29,55 and some less adherent.56 Furthermore, stigma’s consequences may vary for different groups, individuals, and settings.15,42,44,57,58 Intersectional ap- S90 The International Journal of Tuberculosis and Lung Disease Table 2 What are the main drivers and facilitators of TB stigma(s)? No Research questions/ research topics Foundational studies references 1 How do the drivers of TB stigma vary across contexts and among types of stigma? 9,39,47 2 What are the structural drivers of TB stigmas? 39,48 3 What are the drivers and facilitators of compound stigmas in key populations? 24,37,38,49–51 4 How are the drivers of MDR-TB stigma and LTBI stigma different from those of drugsusceptible TB? 52 Level of measurement Individual: patients Health system: HCWs/health managers/CHWs Community: leaders, key spokespersons Structural: policy-level triggers or drivers Societal systems and structures: laws, policies, institutions, rhetorics, architectures Individual: patients Health system: HCWs/health managers/CHWs Community: leaders, key spokespersons TB-affected populations who are also marginalised for other reasons Individual: patients Health system: HCWs/health managers/CHWs Community: leaders, key spokespersons Key TB affected populations Study designs and methodology Qualitative research: ethnography content review Qualitative research: ethnography, policy mapping Ethnography, oral history, indepth interviews Qualitative research: ethnography TB ¼ tuberculosis; HCW ¼ health care worker; CHW ¼ community health worker; MDR-TB ¼ multidrug-resistant TB; LTBI ¼ latent tuberculous infection. proaches that address multiple forms of exclusion are essential.13 Different types of stigma (anticipated/felt, courtesy, internalised/self and enacted) impact at distinct levels (individual, family, community). Building on existing work, we identified the main research questions and gaps in our understanding of the impact of TB stigma(s) (see Table 3). Theme III: What is the global prevalence and distribution of TB stigma(s)? What explains any variation? Policy makers, civil society, research organisations, donors and activists are keen to know where TB stigma is most problematic so that they can channel scarce resources. This theme focuses attention on the macro level: how prevalent is TB stigma across the globe, and how can valid global comparisons be made? (Table 4). Figure 1 Potential impacts of stigma along a person’s TB care itinerary or trajectory (artist: R Takahashi). TB ¼ tuberculosis. Defining the research agenda S91 Table 3 How consequential are TB stigmas? Where are TB stigma(s) impacts felt? No Research questions/research topics Foundational studies references Does TB stigma contribute to poor care seeking or delay in diagnosis? Does stigma hamper quality and completeness of contact and outbreak investigations? Does stigma reduce willingness to disclose TB disease to contacts? Does stigma hamper TB screening/diagnosis? Do health workers hesitate to ask TB patients about other stigmatised identities, diseases or behaviours? How do health workers experience TB stigmas when they provide TB services, does it impact care? Does TB stigma hamper treatment initiation? Does TB stigma(s) worsen TB outcomes via poor adherence or loss to follow-up, death? 59,49,60–64 Symptomatic persons with undiagnosed TB Interviews, life histories 60,63–65 HCWs, persons with TB Record review, key informant interviews (patients and HCWs) 59,33,66,67 Individual: patients Health system: HCWs/health managers/CHWs Community: leaders, key spokespersons 59,33,68 HCWs Interviews, surveys, focus groups 54,69 PTLFU PTLFU surveys 25,56 TB patients who drop out of anti-tuberculosis treatment Relatives of those who have died of suspected TB Affected households Cohort studies, mortality studies, verbal autopsy studies 1 2 3 4 5 6 7 8 9 10 Does stigma contribute to catastrophic costs? Does stigma contribute to the erosion of social capital and social networks (social impacts)? Does stigma hamper full recovery and long-term selfworth (mental health and quality of life)? How consequential are stigmas for public and individual health and human rights? 28 Level of measurement/population Study designs and methodology Macro- and micro-economic analysis Social network studies 70 Health system, community leaders, key spokespeople 24 Current and former TB patients Cohort study; medical record review; surveys (patient, care giver, family) 13,59 Health system; legal and ethics framework Tracking systemic discrimination, harassment, isolation or breaking confidentiality TB ¼ tuberculosis; HCW ¼ health care worker; CHW ¼ community health worker; PTLFU ¼ pre-treatment loss to follow-up. Although normative influences upon stigma have been mapped in broad strokes,16,17,19,45 multi-country studies are few.12,13,32,74 Theme IV: What can be done to reduce the extent and impact of TB stigma(s)? Sommerland et al. report that the strength of the evidence for interventions to reduce TB stigma is limited.11 Well-designed trials to assess the effect of changing societal, community, and individual attitudes and behaviours to reduce stigma(s), as well as interventions maximising resistance, resilience, and strengthening or multiplying allies, are needed.59 Adapting effective interventions designed to address other stigmas, such as leprosy, HIV or mental health, may be an effective way forward. We proposed to build on the stigma-reduction framework of Heijnders and Van der Meij,74 and chose the ‘onion’ as a familiar frame. As TB stigma may operate at all levels from the individual to the community, to health system level and to the wider societal or structural levels (where policies operate), our responses must also target particular social spaces (Figure 2). We recommend that all implementation science specify which drivers, protective factors, actions or behaviours are being targeted and which behavioural, psychological or social theories underpin the intervention. One way of gaining clarity is to insist on determining if an intervention addresses the drivers of TB stigma (fundamental intervention) or if it aims to mitigate its consequences (symptomatic intervention). While both types of interventions may be needed, transparency on where in this chain of ‘driver-to-consequence’ the intervention takes place could help us understand which interventions are working, and why. Practitioners should partner with researchers to evaluate the media materials, curricula and counselling guides that are in use.75 Publishing negative findings is as crucial as documenting success.4,11 Whether or not effective interventions will perform equally well across settings is a research question that must be answered empirically. Rood et al. and S92 The International Journal of Tuberculosis and Lung Disease Table 4 What is the global foot print of TB stigma(s)? What explains this variation? Research questions/research topics Foundational studies references 11 How do we optimise existing TB stigma scales so that they can be used cross-culturally? 10,15,29,25–28,33,36,71 Individual: patients Community Institutions Laws and policies 12 How do we leverage existing survey data? 1,12 Household surveys 13 How do we measure levels of TB stigmas in key population groups? 41–43,47 14 Is it possible to develop valid ‘mini scales’ and ‘rapid assessments’ that capture TB stigma? What explains the variation in stigmas across and within countries? Are there protective factors in some settings? 55,72 Individual: patients Health workers Community: leaders Key populations: men, miners, PLHIV, prisoners, migrants Structural: policy use at national and global level for monitoring and comparing over time Structural: legal frameworks No 15 4,13–15 Level of measurement/population Study designs and methodology Cross-sectional, comparative or longitudinal surveys; prospective observational cohorts; case studies and indepth interviews Secondary analysis of TB nondisclosure for hypothesis generation Based on qualitative work from thematic area 1.2, conduct surveys Embed in existing and continuing surveys, e.g., prevalence surveys; catastrophic cost surveys Ecological analyses Mapping of legal frameworks using adapted legal environment assessments TB ¼ tuberculosis; PLHIV ¼ people living with human immunodeficiency virus. Sommerland et al. argue that as TB stigma is often compounded with other forms of discrimination, combined stigma-reduction efforts are likely to be synergistic (Table 5).11,13 Several behavioural-change theories could be used to inform the design of stigma-reduction efforts in public health. It remains true that some domains of stigma (e.g., reified beliefs, attitudes and behaviours) may be difficult to change, being sustained by powerful interests, habit, culture, history and social determinants. CONCLUSION Given the goal to eliminate suffering in people with TB, and the urgent need to find and treat the 4 million missing patients and strengthen the fight against MDR-TB, there is an increasing mandate for valid methods to estimate the burden of TB stigma(s). One of the main outcomes of the above-mentioned expert meeting in May 2016 was that, while the world wants simple tools and checklists, complex dilemmas remain to be resolved, including clarity on what causes TB stigmas to emerge and thrive in different contexts and populations. The challenge of unpacking and measuring the intersectional aspects of TB stigma, and the low likelihood of a ‘cure’ for universal stigma being found, oblige diverse and innovative approaches. While existing validated scales are useful, new TB stigma scales need to be developed and rigorously validated to fill the gaps. Collectively, the meeting participants believed that a powerful social force such as TB stigma is amenable to appraisal and amelioration. We hope those interested in reducing the suffering due to TB stigma will use the proposed research agenda to join efforts in achieving better measurement of TB stigma with the ultimate goal of developing clear, focused and effective interventions. People affected by TB have a key role to play in the development and implementation of interventions to ensure that science is linked to the reality of their experiences, and that proven strategies for resistance, resilience and countering are funded and fostered. While the research agenda presented will require considerable resources and planning, we are convinced that it is now time to focus on the ‘how’, and not the ‘whether’, to measure and reduce TB stigma. Figure 2 An onion framework: spaces for intervention to reduce stigma.50 Defining the research agenda S93 Table 5 What can be done to reduce TB stigma? (i.e., which interventions work?) No 16 17 Research questions/research topics Does increasing specific kinds of knowledge reduce TB stigma? What is protective? Which legal and policy reforms reduce TB stigma? Foundational studies references Level of measurement/population 13,76 77 Study designs and methodology Pragmatic trials, RCT, stepped wedge designs Structural, e.g., progressive legal interventions for nonadherence; worker privacy for DOT on the job Structural/Health System Medical ethics education/ interpersonal skills/stigma awareness training Repeat legal environment assessment Trend analyses 18 Which educational interventions reduce TB stigma? 32,68 19 Which types of individual-level psychosocial, nutritional, economic supports foster resilience to TB stigma? How can infection control and contact tracing behaviours be modified to reduce stigma? How effective are shifts in language and rhetoric to reduce stigma? 11,36,31,78 Patient-level Household-level Use of simulated patients with students to assess language used with communicating or providing information Assessments of teaching case presentations about people with TB —observe language used Assess the role of peer or health worker support 79 All levels/ultimately structural and discourse levels Pragmatic trials, RCT, stepped wedge designs 80–85 Discourses, laws, media, policies, recording and reporting forms 22 What interventions are effective with those who stigmatise? 32,76,83 Household/family/care givers Policy makers HCW Study using comprehensive critical analysis and discourse analysis of language used; norms present or not present Content review of print and online media to determine how TB is portrayed (e.g., epidemiology, patient names or images) Pragmatic trials, RCT, stepped wedge designs 23 How effective are interventions adapted from other stigma arenas (e.g., mental health, leprosy, HIV)? What interventions are effective against the consequences of stigma? Is stigma reduced by new technology such as video based-DOT/telemedicine and SMS reminders that may increase privacy and confidentiality? 24,77,80,86–88 20 21 24 25 26 27 28 How well do effective stigma reduction interventions translate to other settings? Are some people more resilient to stigma? How can allies be cultivated to combat discrimination? Which interventions are effective against intersectional stigmas? Does improving TB service delivery quality reduce stigma? e.g., Is stigma reduced by rapid diagnostics or shorter regimens? 89,90 Pragmatic trials, RCT, stepped wedge designs Organisational/health system: HCW and managers/CHWs Individual: patient and family Diverse settings Adding new questions to health facility and individual survey questions; qualitative methods looking at individual responses Matched case control study: enrol patients in the interventions not only compare adherence and health outcomes to nonintervention groups, but ask questions about extent of stigma and shame Multisite studies 12,91,92 Patient-level Case control Participant observation 37,59 Patient-level RCT Pragmatic trials 32 Facility-level Nested studies in evaluations of non-stigma interventions Pragmatic trials TB ¼ tuberculosis; RCT ¼randomised controlled trial; HCW ¼ health care worker; HIV ¼ human immunodeficiency virus; DOT ¼ directly observed treatment; SMS ¼ short message service; CHW ¼ community health worker. S94 The International Journal of Tuberculosis and Lung Disease Acknowledgements We are indebted to the anonymous reviewers for constructive comments. The Global Health Bureau, Office of Health, Infectious Disease and Nutrition (HIDN), US Agency for International Development (USAID, Washington DC, USA), financially supports this study through Challenge TB under the terms of Agreement No. AIDOAA-A-14-00029. The authors’ views expressed in this publication do not necessarily reflect the views of the USAID or US Government. Conflicts of interest: none declared. 18 19 20 21 References 1 World Health Organization, United Nations. Joint United Nations statement on ending discrimination in health care settings. Geneva, Switzerland: WHO, 2017. http://www.who. int/mediacentre/news/statements/2017/discrimination-inhealth-care/en/ Accessed August 2017. 2 World Health Organization. The End TB Strategy. Geneva, Switzerland: WHO, 2015. 3 Coleman C H, Jaramillo E, Reis A, Selgelid M. Guidance on ethics of tuberculosis prevention, care and control. Geneva, Switzerland: World Health Organization, 2010: p 38. 4 Oxley A. The international TB response: what are we missing? Lancet Glob Health Blog 2014. http://globalhealth.thelancet. com/2014/03/24/international-tb-response-what-are-wemissing Accessed August 2017. 5 World Health Organization. End TB Strategy. Geneva, Switzerland: WHO, 2014. 6 Uys L, Chirwa M, Kohi T, et al. Evaluation of a health settingbased stigma intervention in five African countries. AIDS Patient Care STDS 2009; 23: 1059–1066. 7 Oldekop J A, Fontana L B, Grugel J, et al. 100 key research questions for the post-2015 development agenda. Dev Policy Rev 2016; 34: 55–82. 8 United Nations. Equality and non-discrimination at the health of sustainable development: a shared united nations framework for action. Vol 20183. New York, NY, USA: UN, 2016. 9 Weiss M G, Ramakrishna J, Somma D. Health-related stigma: rethinking concepts and interventions. Psychol Health Med 2006; 11: 277–287. 10 Kipp A M, Ewing H, Redwood L, Mitchell E M H. Overview of the TB stigma scale landscape: what do the validation studies tell us about the utility of TB stigma scales? Presented at the TB Stigma Measurement Challenge Expert Consultation, The Hague, Netherlands, 17–18 May 2016 11 Sommerland N, Wouters E, Mitchell E M H, et al. Interventions to reduce tuberculosis stigma: a systematic review. Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S81–S86. 12 Straetemans M, Bakker M I, Mitchell E M H. Correlates of observing and willingness to report stigma towards HIV clients by (TB) health workers in Africa. Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S6–S18. 13 Rood E J J, Mergenthaler C, Bakker M I, Redwood L, Mitchell E M H. Using 15 DHS surveys to study epidemiological correlates of TB courtesy stigma and health-seeking behaviour. Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S60–S68. 14 Chang S-H, Cataldo J K. A systematic review of global cultural variations in knowledge, attitudes and health responses to tuberculosis stigma. Int J Tuberc Lung Dis 2014; 18: 168–173. 15 Somma D, Gosoniu G D, Ganapathy S, et al. Gender and sociocultural determinants of delay to diagnosis of TB in Bangladesh, India and Malawi. Int J Tuberc Lung Dis 2008; 12: 848–855. 16 Pescosolido B A. The stigma complex. Annu Rev Sociol 2015; 41: 87–116. 17 Pescosolido B A, Martin J K, Lang A, Olafsdottir S. Rethinking theoretical approaches to stigma: a framework integrating 22 23 24 25 26 27 28 29 30 31 32 33 34 35 normative influences on stigma (FINIS). Soc Sci Med 2008; 67: 431–440. Van Brakel W. Measuring health-related stigma: a literature review. Psychol Health Med 2006; 11: 307–334. Link B G, Phelan J C. Conceptualizing stigma. Annu Rev Sociol 2001; 27: 363–385. Stevelink S A M, van Brakel W H, Augustine V. Stigma and social participation in Southern India: Differences and commonalities among persons affected by leprosy and persons living with HIV/AIDS. Psychol Health Med 2011; 16: 695–707. Livingston J D, Boyd J E. Correlates and consequences of internalized stigma for people living with mental illness: a systematic review and meta-analysis. Soc Sci Med 2010; 71: 2150–2161. Goffman E. Stigma: notes on the management of spoiled identity. Englewood Cliffs, NJ, USA: Prentice-Hall, 1963. Mitchell E M H. The masks we wear: authenticity, vulnerability, and innovation in TB. Int J Tuberc Lung Dis 2017; 21: 127–2017. France N F, Macdonald S H, Conroy R R, et al. ‘An unspoken world of unspoken things’: A study identifying and exploring core beliefs underlying self-stigma among people living with HIV and AIDS in Ireland. Swiss Med Wkly 2015; 145: w14113. Kipp A M, Pungrassami P, Nilmanat K, et al. Sociodemographic and AIDS-related factors associated with tuberculosis stigma in southern Thailand: a quantitative, cross-sectional study of stigma among patients with TB and healthy community members. BMC Public Health 2011; 11: 675. Van Rie A, Sengupta S, Pungrassami P, et al. Measuring stigma associated with tuberculosis and HIV/AIDS in southern Thailand: exploratory and confirmatory factor analyses of two new scales. Trop Med Int Health 2008; 13: 21–30. Jaramillo E. Tuberculosis and stigma: predictors of prejudice against people with tuberculosis. J Health Psychol 1999; 4: 71– 79. Colvin C. Symptoms, stigma, and strikes: an exploration of factors related to delayed diagnosis of tuberculosis in San Pedro Sula, Honduras. Tulane University New Orleans, LA, USA: Digital Library, 2005. https://digitallibrary.tulane.edu/ islandora/object/tulane%3A26135 Accessed August 2017. Kipp A M, Pungrassami P, Stewart P W, Chongsuvivatwong V, Strauss R P, Van Rie A. Study of tuberculosis and AIDS stigma as barriers to tuberculosis treatment adherence using validated stigma scales. Int J Tuberc Lung Dis 2011; 15: 1540–1546. Jittimanee S X, Nateniyom S, Kittikraisak W, et al. Social stigma and knowledge of tuberculosis and HIV among patients with both diseases in Thailand. PLOS ONE 2009; 4: e6360. Macq J, Solis A, Martinez G, Martiny P. Tackling tuberculosis patients’ internalized social stigma through patient centred care: an intervention study in rural Nicaragua. BMC Public Health 2008; 8: 154. Bond V, Floyd S, Fenty J, et al. Secondary analysis of TB stigma data from a cluster randomised trial in Zambia and South Africa (ZAMSTAR). Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S49–S59. Wouters E, Rau A, Engelbrecht M, et al. The development and piloting of parallel scales measuring external and internal HIV and tuberculosis stigma among healthcare workers in the Free State Province, South Africa. Clin Infect Dis 2016; 62 (Suppl 3): S244–S254. Courtwright A, Turner A N. Tuberculosis and stigmatization: pathways and interventions. Public Health Rep 2010; 125 (Suppl 4): 34–42. Dodor E A. An exploration of the causes, manifestations and consequences of tuberculosis stigma in an urban district in Ghana. Int J Tuberc Lung Dis 2009; 12: 1048–1054. Defining the research agenda 36 Macq J, Solis A, Martinez G. Assessing the stigma of tuberculosis. Psychol Health Med 2006; 11: 346–352. 37 Hayes-Larson E, Hirsh-Moverman Y, Saito S, et al. High baseline prevalence of stigma, depressive symptoms and hazardous alcohol use among TB-HIV patients in Lesotho. Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S34–S41. 38 Møller V, Erstad I, Zani D. Drinking, smoking, and morality: do ’drinkers and smokers’ constitute a stigmatised stereotype or a real TB risk factor in the time of HIV/AIDS? Soc Indic Res 2010; 98: 217–238. 39 Coreil J, Mayard G, Simpson K M, Lauzardo M, Zhu Y, Weiss M. Structural forces and the production of TB-related stigma among Haitians in two contexts. Soc Sci Med 2010; 71: 1409– 1417. 40 Juniarti N, Evans D. A qualitative review: the stigma of tuberculosis. J Clin Nurs 2011; 20: 1961–1970. 41 Bond V, Nyblade L. The importance of addressing the unfolding TB-HIV stigma in high HIV prevalence settings. J Community Appl Soc Psychol 2006; 16: 452–461. 42 Daftary A. HIV and tuberculosis: the construction and management of double stigma. Soc Sci Med 2012; 74: 1512– 1519. 43 Craig G M, Daftary A, Engel N, O’Driscoll S, Ioannaki A. Tuberculosis stigma as a social determinant of health: a systematic mapping review of research in low incidence countries. Int J Infect Dis 2017; 56: 90–100. 44 Karim F, Chowdhury A M R, Islam A, Weiss M G. Stigma, gender, and their impact on patients with tuberculosis in rural Bangladesh. Anthropol Med 2007; 14: 139–151. 45 Pescosolido B A, Martin J K, Lang A, et al. Stigma and its public health implications. Soc Sci Med 2006; 367: 1–28. 46 Thomas B E, Shanmugam P, Malaisamy M, et al. Psychosocioeconomic issues challenging multidrug resistant tuberculosis patients: a systematic review. PLOS ONE 2016; 11: e0147397. 47 Baral S C, Karki D K, Newell J N. Causes of stigma and discrimination associated with tuberculosis in Nepal: a qualitative study. BMC Public Health 2007; 7: 211. 48 Farmer P, Robin S, Ramilus S L, Kim J Y. Tuberculosis, poverty, and ‘compliance’: lessons from rural Haiti. Semin Respir Infect 1991; 6: 254–260. 49 Chikovore J, Hart G, Kumwenda M, Chipungu G A, Desmond N, Corbett L. Control, struggle, and emergent masculinities: a qualitative study of men’s care-seeking determinants for chronic cough and tuberculosis symptoms in Blantyre, Malawi. BMC Public Health 2014; 14: 1053. 50 Donahue M C, Dube Q, Dow A, Umar E, Van Rie A. ‘They have already thrown away their chicken’: barriers affecting participation by HIV-infected women in care and treatment programs for their infants in Blantyre, Malawi. AIDS Care 2012; 24: 1233–1239. 51 Møller V, Erstad I, Cramm J M, et al. Delays in presenting for tuberculosis treatment associated with fear of learning one is HIV-positive. Afr J AIDS Res 2011; 10: 25–36. 52 Daftary A, Padayatchi N. Provider perspectives on drugresistant tuberculosis and human immunodeficiency virus care in South Africa: a qualitative case study. Int J Tuberc Lung Dis 2016; 20: 1483–1488. 53 Kipp A M. Overview of the TB stigma scale landscape: validity, utility and robustness of existing measures for use in measuring progress in reducing TB stigma. 47th Union Conference on Lung Health. Liverpool, UK, 26–29 October 2016. Int Tuberc Lung Dis 2016; 20 (Suppl 1): S25. 54 Murray E J, Bond V A, Marais B J, Godfrey-Faussett P, Ayles H M, Beyers N. High levels of vulnerability and anticipated stigma reduce the impetus for tuberculosis diagnosis in Cape Town, South Africa. Health Policy Plan 2013; 28: 410–418. 55 Cremers A L, Gerrets R, Kapata N, et al. Tuberculosis patients’ pre-hospital delay and non-compliance with a longstanding 56 57 58 59 60 61 62 63 64 65 66 67 68 69 70 71 72 S95 DOT programme: a mixed methods study in urban Zambia. BMC Public Health 2016; 16: 1130. Munro S A, Lewin S A, Smith H J, Engel M E, Fretheim A, Volmink J. Patient adherence to tuberculosis treatment: a systematic review of qualitative research. PLOS Med 2007; 4: e238. Chikovore J, Hart G, Kumwenda M, Chipungu G A, Desmond N, Corbett E L. TB and HIV stigma compounded by threatened masculinity: the implications for tuberculosis health careseeking in Malawi. Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S26–S33. Miller C, Huston J, Samu L, Mfinanga S, Hopewell P, Fair E. ‘It makes the patient’s spirit weaker’: tuberculosis stigma in Dar es Salaam, Tanzania. Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S42–S48. Sommerland N, Wouters E, Masquillier C, et al. Stigma as a barrier to the use of occupational health units for TB services in South Africa. Int J Tuberc Lung Dis 2017; 21 (Suppl 1): S75– S80. Faccini M, Cantoni S, Ciconali G, et al. Tuberculosis-related stigma leading to an incomplete contact investigation in a lowincidence country. Epidemiol Infect 2015; 143: 2841–2848. Mulder C, Harting J, Jansen N, Borgdorff M W, van Leth F. Adherence by Dutch public health nurses to the national guidelines for tuberculosis contact investigation. PLOS ONE 2012; 7: e49649. Cook V J, Sun S J, Tapia J, et al. Transmission network analysis in tuberculosis contact investigations. J Infect Dis 2007; 196: 1517–1527. Klovdahl A S, Graviss E A, Yaganehdoost A, et al. Networks and tuberculosis: an undetected community outbreak involving public places. Soc Sci Med 2001; 52: 681–694. Yaganehdoost A, Graviss E A, Ross M W, et al. Complex transmission dynamics of clonally related virulent Mycobacterium tuberculosis associated with barhopping by predominantly human immunodeficiency virus–positive gay men. J Infect Dis 1999; 180: 1245–1251. Centers for Disease Control and Prevention. HIV-related tuberculosis in a transgender Network—Baltimore, Maryland, and New York City Area, 1998–2000. JAMA 2000; 283: 2515–2516. Skinner D, Claassens M M. It’s complicated: why do tuberculosis patients not initiate treatment? A qualitative study from South Africa. BMC Infect Dis 2016; 16: 712. Dodor E A, Kelly S, Neal K. Health professionals as stigmatisers of tuberculosis: Insights from community members and patients with TB in an urban district in Ghana. Psychol Health Med 2009; 14: 301–310. Buregyeya E, Nuwaha F, Wanyenze R K, et al. Utilization of HIV and tuberculosis services by health care workers in Uganda: implications for occupational health policies and implementation. PLOS ONE 2012; 7: e46069. MacPherson P, Houben R M G J, Glynn J R, Corbett E L, Kranzer K. Pre-treatment loss to follow-up in tuberculosis patients in low- and lower-middle-income countries and highburden countries: a systematic review and meta-analysis. Bull World Health Organ 2014; 92: 126–138. Kaawa-Mafigiri D. Social networks and social support for tuberculosis control in Kampala, Uganda. Cleveland, OH, USA: Case Western Reserve University, 2007. Atre S R, Kudale A M, Morankar S N, Rangan S G, Weiss M G. Cultural concepts of tuberculosis and gender among the general population without tuberculosis in rural Maharashtra, India. Trop Med Int Health 2004; 9: 1228–1238. Colvin C. Is attitude toward TB disease disclosure a valid proxy measure of anticipated TB stigma in the general population? Evidence from Honduras. 47th Union Conference on Lung Health, Liverpool, UK, 26–29 October, 2016. Int J Tuberc Lung Dis 2016: 20 (Suppl 1): S26. S96 The International Journal of Tuberculosis and Lung Disease 73 Somma D, Thomas B E, Karim F, et al. Gender and sociocultural determinants of TB-related stigma in Bangladesh, India, Malawi and Colombia. Int J Tuberc Lung Dis 2008; 12: 856–866. 74 Heijnders M, Van Der Meij S. The fight against stigma: an overview of stigma-reduction strategies and interventions. Psychol Health Med 2006; 11: 353–363. 75 Smith R A. Media depictions of health topics: challenge and stigma formats. J Health Commun 2007; 12: 233–249. 76 Balogun M, Sekoni A, Meloni S T, et al. Trained community volunteers improve tuberculosis knowledge and attitudes among adults in a periurban community in southwest Nigeria. Am J Trop Med Hyg 2015; 92: 625–632. 77 Stangl A L, Lloyd J K, Brady L M, Holland C E, Baral S. A systematic review of interventions to reduce HIV-related stigma and discrimination from 2002 to 2013: how far have we come? J Int AIDS Soc 2013; 16: 18734. 78 van Hoorn R, Jaramillo E, Collins D, Gebhard A, van den Hof S. The effects of psycho-emotional and socio-economic support for tuberculosis patients on treatment adherence and treatment outcomes – a systematic review and meta-analysis. PLOS ONE 2016; 11: e0154095. 79 Buregyeya E, Mitchell E M H, Rutebemberwa E, et al. Acceptability of masking and patient separation to control nosocomial Tuberculosis in Uganda: a qualitative study. J Public Health 2012; 20: 599–606. 80 Clair M, Daniel C, Lamont M. Destigmatization and health: cultural constructions and the long-term reduction of stigma. Soc Sci Med 2016; 165: 223–232. 81 Corrigan P W. Best practices: Strategic stigma change (SSC): five principles for social marketing campaigns to reduce stigma. Psychiatr Serv 2011; 62: 4–6. 82 Smith R A. Testing the model of stigma communication with a factorial experiment in an interpersonal context communication with a factorial experiment in an interpersonal context. Commun Stud 2014; 65: 154–173. 83 Smith R A. An experimental test of stigma communication content with a hypothetical infectious disease alert an experimental test of stigma communication content with a hypothetical infectious disease alert. 2017; 84 Smith R A, Ferrara M, Witte K. Social sides of health risks: stigma and collective efficacy social sides of health risks. Health Commun 2007; 21: 55–64. 85 Soffer M. Illness-related stigma in the Israeli Press: a study of media coverage of severe physical illnesses. Stigma Res Action 2011; 2: 28–33. 86 Peters R M, Dadun, Zweekhorst M B, Bunders J F, Irwanto, van Brakel W H. A cluster-randomized controlled intervention study to assess the effect of a contact intervention in reducing leprosy-related stigma in Indonesia. PLOS Negl Trop Dis 2015; 9: e0004003. 87 McGinty E E, Goldman H H, Pescosolido B, Barry C L. Portraying mental illness and drug addiction as treatable health conditions: effects of a randomized experiment on stigma and discrimination. Soc Sci Med 2015; 126: 73–85. 88 Corrigan P W, Shapiro J R. Measuring the impact of programs that challenge the public stigma of mental illness. Clin Psychol Rev 2010; 30: 907–922. 89 Corneli A, Jarrett N M, Sabue M, et al. Patient and provider perspectives on implementation models of HIV counseling and testing for patients with TB. Int J Tuberc Lung Dis 12 (Suppl 1): 79–84. 90 Nabbuye-Sekandi J, Okot-Chono R, Rusen I D, et al. Factors associated with human immunodeficiency virus testing among tuberculosis patients receiving treatment at health facilities in Uganda. Int J Tuberc Lung Dis 2010; 14: 896–902. 91 Ashforth B E, Kreiner G E. Dirty work and dirtier work: differences in countering physical, social, and moral stigma. Manag Organ Rev 2014; 10: 81–108. 92 Kreiner G E, Ashforth B E, Sluss D M. Identity dynamics in occupational dirty work: integrating social identity and system justification perspectives. Organ Sci 2006; 17: 619–636. Defining the research agenda i R É S U M É La stigmatisation vis-à-vis de la tuberculose (TB) reçoit enfin, au niveau mondial, une attention bien méritée, car elle est cruciale pour identifier et traiter les 4 millions de patients atteints de tuberculose (TB) qui sont actuellement manqués par les programmes nationaux TB. La capacité à mesurer et à évaluer le succès des efforts de réduction de la stigmatisation TB est cependant limit ée par le besoin d’outils supplémentaires. Lors d’une réunion de mesure de la stigmatisation TB qui a eu lieu à La Haye en 2016, les experts en stigmatisation ont discuté et proposé un programme de recherche autour de quatre thèmes : 1) Moteurs : quels sont les principaux moteurs et domaines de la stigmatisation TB ? 2) Conséquences : quelles sont les conséquences de la stigmatisation TB? Comment les impacts négatifs sont-ils surtout ressentis? 3) Fardeau : quelle est la prévalence mondiale et la distribution de la stigmatisation TB? Comment s’expliquent les variations? 4) Intervention : que peut-on faire pour réduire l’étendue et l’impact de la stigmatisation TB? Chaque thème a ensuite été subdivisé en sujets de recherche à examiner pour faire avancer les choses. Ceci inclut de clarifier les causes qui font émerger et croı̂tre la stigmatisation vis-à-vis de la TB, la difficulté à mesurer la complexité de la stigmatisation et l’absence de probabilité d’un remède universel. Néanmoins, ces défis ne devraient pas entraver les investissements dans la mesure et dans la réduction de la stigmatisation relative à la TB. Nous pensons qu’il est temps pour la communauté mondiale de se concentrer sur la manière de mesurer et de réduire la stigmatisation liée à la TB et non pas de se demander s’il faut le faire. RESUMEN Dada la gran influencia que ejercen los estigmas relacionados con la tuberculosis (TB) en la detección y el tratamiento de los 4 millones de pacientes que se pasan por alto en los programas nacionales contra la TB, se otorga hoy a la estigmatización una atención bien justificada y tardı́a a escala mundial. Sin embargo, la falta de instrumentos adecuados limita la capacidad de medir y evaluar la eficacia de las iniciativas tendentes a aminorar los estigmas asociados con la TB. En una reunión celebrada en el 2016 en La Haya sobre la medición de la estigmatización por TB, expertos en el tema analizaron y propusieron un programa de investigación en torno a los siguientes cuatro temas: 1) los factores determinantes: ¿Cuáles son los principales factores y las dimensiones de la estigmatización por TB?; 2) las consecuencias: ¿Qué tanta repercusión tienen los estigmas relacionados con la TB? ¿De qué manera se suelen percibir las repercusiones negativas?; 3) la carga: ¿Cuál es la prevalencia mundial de estigmatización por tuberculosis y cómo se distribuye? ¿Cómo se explican las variaciones? 4): la intervención: ¿Qué puede hacerse a fin de disminuir la magnitud y el impacto de la estigmatizaci ón por TB? Luego, cada tema se subdividió en los aspectos de investigación que se deben abordar con el fin de avanzar en el programa. Estos aspectos incluyeron una mayor claridad sobre las causas que hacen surgir y progresar los estigmas relacionados con la TB, la dificultad de medir la complejidad de los estigmas y la improbabilidad de un ‘remedio’ universal a la estigmatización. Sin embargo, estas dificultades no deberı́an obstaculizar las inversiones encaminadas a medir los estigmas relacionados con la TB y a reducirlos. Los autores consideran que llegó el momento de centrar los esfuerzos en decidir la forma como la comunidad mundial debe medir y reducir la estigmatización por TB en lugar de deliberar sobre la pertinencia de hacerlo.