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What Do We Know About Medical Tourism? A Review of the Literature With Discussion of Its Implications for the UK National Health Service as an Example of a Public Health Care System

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Johanna Hanefeld, Richard Smith, Daniel Horsfall, Neil Lunt, What Do We Know About Medical Tourism? A Review of the Literature With Discussion of Its Implications for the UK National Health Service as an Example of a Public Health Care System, Journal of Travel Medicine , Volume 21, Issue 6, 1 November 2014, Pages 410–417, https://doi.org/10.1111/jtm.12147

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Medical tourism is a growing phenomenon. This review of the literature maps current knowledge and discusses findings with reference to the UK National Health Service (NHS).

Databases were systematically searched between September 2011 and March 2012 and 100 papers were selected for review.

The literature shows specific types of tourism depending on treatment, eg, dentistry, cosmetic, or fertility. Patient motivation is complex and while further research is needed, factors beyond cost, including availability and distance, are clearly important. The provision of medical tourism varies. Volume of patient travel, economic cost and benefit were established for 13 countries. It highlights contributions not only to recipient countries' economies but also to a possible growth in health systems' inequities. Evidence suggests that UK patients travel abroad to receive treatment, complications arise and are treated by the NHS, indicating costs from medical travel for originating health systems.

It demonstrates the importance of quality standards and holds lessons as the UK and other EU countries implement the EU Directive on cross‐border care. Lifting the private‐patient‐cap for NHS hospitals increases potential for growth in inbound medical tourism; yet no research exists on this. Research is required on volume, cost, patient motivation, industry, and on long‐term health outcomes in medical tourists.

Medical tourism—people traveling abroad with the expressed purpose of accessing medical treatment—is a growing phenomenon associated with globalization. 1 This includes cheaper and more widely available air travel and cross‐border communication through the Internet, which allows medical providers from one country to market themselves to patients in another. 2 At the same time, increased movement of health workers for education means doctors providing care in middle‐ and low‐income countries have in many cases the same qualifications as those in the high‐income countries in Western Europe and the United States. This has been coupled with an increase in foreign direct investment in health care providers in destination countries. 3 The increasing acceptance of health care portability is evident in Europe where greater patient mobility led to an EU Directive on cross‐border health care. 4 Together with a rise in out‐of‐pocket expenditures for health in many high‐income countries at a time of economic crisis, this conspires to form a perfect storm for medical tourism.

Yet, understanding of medical travel is limited. 5 Little is known as to which patients choose to travel and why, when others do not. Details of the volume of patient flows and resources spent remain uncertain. 3 This has hampered efforts to understand the economic costs and benefits to countries experiencing inflows and outflows of patients. Similarly, for the medical tourism industry, the role of private providers and brokers and marketing remain a “black box.” 1 While interest in the issue has grown over the past decade, effects on patients and health systems are not fully understood.

This review of the literature seeks to outline the current level of knowledge on medical tourism. Specifically, it aims to better understand (1) patient motivation, (2) the medical tourism industry, (3) volume of medical travel, and (4) effects of medical travel on originating health systems. Results are reported and discussed, paying specific attention to evidence of impact and lessons for the UK National Health Service (NHS) as an example of how medical tourism affects even universal public health systems. The authors conclude on current levels of knowledge, critical gaps, and future research priorities on medical travel.

The review was conducted between September 2011 and March 2012 as part of wider research, assessing implications of medical tourism on the UK NHS. Authors developed a search strategy based on the aims set out above. They adapted the strategy used by Smith and colleagues, 5 deemed particularly relevant as it presented a recent review of medical tourism albeit focused on bilateral tourism. It was amended to focus more broadly on medical tourism. Initial papers identified were reviewed for inclusion by J. H. and R. S. according to title and where this proved inconclusive according to the abstract. In line with research objectives, papers with general focus on medical tourism, published in English and German (languages read by authors), and focused on the NHS, were included. The following were excluded: papers on well‐being, news items, commentaries, laws or directives, and conference proceedings; papers focusing on stem cell tourism, travel for assisted suicide, and transplant tourism, given the distinct ethical issues. Three hundred and seventy‐four papers remained as initial sample. References of papers identified were further examined to ensure comprehensiveness and four additional papers were included. The initial selection of papers was then reviewed (abstract or full paper) applying these criteria and focusing more specifically on the aims of the review (as above). Two papers were not accessible and therefore excluded. 6,7 A final list of 100 papers was derived for inclusion in this review. This sample was tested based on the criteria by D. H. The literature search is summarized in the PRISMA flowchart (Figure 1 ).

PRISMA flow diagram for literature review medical tourism.

A rapidly expanding literature over the past 5 years with an “explosion” in 2010 and 2011 is reflected in the dates of publication of papers included in the review—73 were published in 2010 and 2011.

Types of Studies Reviewed

Papers included in the review were classified into the following categories: (1) those based on primary data collection (quantitative and qualitative): interviews, surveys, analysis of datasets collected and obtained by authors, or the calculation of revenue and tourist flows, and case studies of patients; (2) reviews: literature reviews of medical tourism websites or promotional materials; (3) analysis: papers which while drawing on secondary sources, provide substantive new insights or conceptualize it in a new way (a number of papers presented frameworks); and (4) overview articles which gave an introduction to the issue of medical tourism. The results are summarized in Figure 2 .

Type of study reviewed.

Geographic Focus

Papers were grouped according to the region the research investigated (see Figure 3 ), or global where they were general. Europe was the focus of 29 papers, 13 explicitly focus on the UK and a further 11 papers refer to either UK patients or the NHS, thus a total of 24 papers mentioned or focused on the UK.

Countries covered.

Literature reviewed suggests a regional dimension to medical tourism: Japanese companies send their employees to Thailand, 8 or to countries in the Gulf. 9,10 A study of medical tourists in Tunisia found that they were from neighboring countries. 11 Countries are known for specific areas of medicine: Singapore for high‐end procedures, 12 Thailand for cardiac, orthopedic, and gender reassignment surgery, 13 Eastern Europe for dental tourism, 14 and Spain for fertility treatment. 15 While some destinations were recognized as popular with UK patients, eg, Budapest for dental treatment, evidence from literature suggested that proximity alone does explain preference for one destination over another.

Motivation to Travel

Most papers made reference to push and pull factors determining patients' decision to travel. These relate to cost, perceived quality, familiarity, waiting lists or delays in treatment, or the lack of availability of certain treatments in the country of origin. 16 As this list demonstrates, these are often complex and dynamic, 6 and may vary according to the treatment for which a patient travels. Evidence suggests that patients traveling for cosmetic surgery may enjoy the anonymity of a destination far from their country of origin, 17 whereas migrants may prefer to return “home” to feel more comfortable with language or type of care provided. 18 These different factors allow for a division into different subsets of medical tourism.

A number of studies refer to a group of tourists classified as diaspora, documenting the return of recent migrants from India, China, Korea, and Mexico, to access treatment either not available or perceived to be not available in their country of residence, or perceived to be more effective. 9,18–20 While cost plays a part in explaining why, eg, Mexican immigrants to the United States return to Mexico for treatment, trust emerged as the key determining factor. This may partly be linked to language barriers, as a study of Korean immigrants to Australia suggests. 18

Reproductive or fertility travel is comparatively better documented than other forms of medical tourism. 15 Of the 16 papers identified for inclusion in this review, 6 papers focus on equity and ethical issues relating to fertility tourism, highlighting the rights of women in recipient countries and equity concerns where they may be compelled by poverty to donate eggs or act as surrogates. Four papers provide a general overview of the issues relating to fertility tourism. 7,21–23 A review of literature on cross‐border reproductive care 15 finds a consistent gap of empirical research—of 54 papers reviewed only 15 were based on empirical investigation. It noted the absence of evidence about patients' backgrounds and factors motivating their travel, and a gap in research on industry. A specific feature of fertility travel cited across papers reviewed is that availability of treatment (in this case gametes and surrogacy) is a factor in patient decision making. This includes the wish for timely and affordable treatment; in the UK it includes perceptions of the NHS as stressful and less effective. 6 Evidence also highlights health effects of fertility travel on patients, showing an increase in multiple births in a London hospital resulting from fertility treatment received abroad. 24 Combined, these studies show that there is an effect of fertility travel on the health system of the country from which medical tourists originate, in this case the NHS, and that regulation of availability and (perceived) quality of service are factors leading patients to travel.

Dental Tourism, Bariatric and Cosmetic Surgery

Other types of tourism are identifiable, including dental tourism. 25 Three papers 26,27,28 indicated this is likely to be an area of increasing travel by UK citizens, given the high cost of dentistry in the UK private sector, limited availability in the public sector, and lower cost in Eastern Europe. 27 A survey of dental clinics in Western Hungary and Budapest showed the largest group of patients (20.2%) originating from the UK with lower prices cited as main motivating factor. 28 Two papers focused in depth on issues surrounding bariatric surgery, exploring the ethical challenges and a case study of complications experienced by a US patient. 29,30 Papers by Birch and colleagues 31 and Miyagi and colleagues 32 focus on complications from cosmetic tourism in UK patients. Others reported that a poll conducted amongst the members of the UK public found that 92% would consider traveling abroad for cosmetic surgery. 33 The possibility of a large number of UK patients seeking cosmetic surgery abroad appears supported by a survey conducted by the British Association of Plastic, Reconstructive and Aesthetic Surgeons which found that 37% of respondents had seen patients in the NHS with complications from overseas surgery. 31

Risks for patients are covered in 29 papers. But surprisingly only 8 of these papers focus exclusively on the issue, and 10 studies mention longer‐term health outcomes of patients. Three describe the recent outbreak of NDM1 bacteria following patients receiving treatment in India, a fourth describes an outbreak of hepatitis B in a London hospital traced to a patient recently returned from surgery in India, pointing to potential risks of diaspora travel. 34–37

While papers tend to mention regulation, only two 38,39 review this more systematically. Both point to a vacuum in regulation, with no one specific regulator or quality assurance standard in place, but rather a number of private companies offering quality assurance through affiliation, creating a market for quality assurance rather than independent standards.

Effect on Countries

As summarized in Table 1 , 37 papers focused on the effects on recipient country's health system. Issues highlighted include the potential for medical tourism to retain or attract doctors in low‐ and middle‐income countries who may otherwise emigrate, thus preventing or reversing a brain drain, and generating foreign currency. 12 Also considered is the danger of creating a two‐tiered health system, resulting in increasing inequities in access and quality of health care for the local population in destination countries, 40,41 mainly as a result of a rise in price where public health services are not provided for free in recipient countries, and the potentially greater concentration of doctors in the private sector. 42

Issues covered

A total of 34 papers focused on potential effects on originating countries' health system. These referred to factors leading to patients' travel, including rise in costs. Papers documented patients returning with complications. 43 Seven papers specifically highlighted complications dealt with in the NHS. 31 Research highlighted the need for regulation, the lack of quality control of overseas providers, and the cost (potential or real) arising to the originating country from treating such complications. Two papers calculated the potential cost saving and benefits of sending patients abroad. 20,44 Overall, papers focusing on the effects on originating countries' health system concentrate mainly on perceived negative consequences.

Forty‐one papers reviewed focus at least partly on providers of medical tourism. A subset of 22 papers studied the medical tourism industry in a more focused way. These provide evidence of a highly diversified industry, with no clear typology emerging. For example, in Southeast Asia medical tourism is state‐led, with large hospitals targeting foreign patients. In other cases, such as cosmetic or dental tourism, intermediaries organize travel and treatment for patients. Examining the entire literature, it is clear that there is not a uniform model or chain for medical tourism.

Articles examining communication materials and websites highlight the limited information on follow‐up care and redress in case of complications. 2 They point to an emphasis on testimonies by patients, rather than formal accreditation or qualification of clinicians, a focus on tourism aspects of the destination and on trust—offering services “as good as at home.” 41 These are in addition to low cost used as a selling point. Studies focusing on medical tourism facilitators identify these as a heterogeneous group. 45,46

Papers reviewed mention individual hospitals or a medical tourism provider at the country level to give a flavor of the industry. 8,45 However, only four papers 47–50 report findings of a more systematic assessment of the industry, including focus on the strong state role in the development of medical tourism in Hong Kong, Malaysia, and Singapore, analyzing how these countries have fostered medical tourism, including through tax incentives. Singapore, for example, made a conscious decision to focus on the high‐end complex procedures to have a competitive advantage. 47

Number of People Traveling

The actual volume in flow of medical patients was referred to in many papers but investigated in few 10,11,28,42,51–54 ; all papers provided further estimates or trends. Most papers cited similar figures of patient flows, but often sources were not accessible or based on media reports or other academic papers, which in turn quoted inaccessible sources. Seven papers referred directly to a report by Deloitte Consultancy, and six to McKinsey; the exact ways in which these were calculated remain unclear. Even where these were not referenced, the figures cited suggest these two reports as a source. For example, one paper 33 cites The Economist stating 750,000 US patients traveling abroad for treatment in 2007. This is the figure provided in the report from Deloitte consultancy in 2008.

Eight papers reviewed had either generated or collected own data on patient flows. Only three papers had calculated the total volume of medical tourism for 13 countries, including actual cost and effect on recipient country's health systems. NaRanong and colleagues calculate the contribution of medical tourism to Thai GDP (0.4%), while medical tourists with their higher purchasing power are likely to increase the cost of health services and lessen access in the public sector. 42 This contrasts with Lautier's findings which highlight that export of health services in Tunisia simply makes use of excess capacity in the country's private sector. 11 Siddiqi's 11‐country study in the Middle East showed complex flows within the region. 10 Findings across the different studies suggest that the impact on recipient country's health system depends on the context and capacity, but that there is likely to be a small contribution to overall GDP. How income gained from medical tourism is in turn invested has not been studied.

This review of the literature provides the most comprehensive overview of knowledge on medical tourism to date. The main limitation of the studies is the focus on English (and German) literature, and as set out in the search strategy a narrow definition focused on medical tourism rather than on the inclusion of broader health and well‐being travel literature. This was essential to maintain feasibility of the review, given the large number of papers published.

Main Findings: What We Can Learn From the Literature on Medical Tourism

The literature reviewed clearly indicates that medical tourism is no unified phenomenon. Sub‐types of travel, such as diaspora or fertility travel, travel for bariatric surgery, dental, or cosmetic work, were evident from the review. Decisions by patients to travel are not simply guided by cost considerations or even clinical outcomes. Rather, the literature points to a complex matrix of perceptions of care, waiting times, cost, and others, depending on the type of treatment sought. For example, trust appeared as a motivation for diaspora travelers but not for dental tourists, cost or availability in cosmetic procedures, and regulation in the case of fertility. However, lack of information about patients' characteristics limits deeper understanding of push and pull factors. With very few exceptions, 16,6 the absence of in‐depth interviews with more than one or two patients poses the greatest weakness of the literature reviewed and opens the potential to bias within studies reviewed.

A diverse picture of the medical tourism industry emerges. Some countries have become known for excellence in certain areas of treatment such as Spain for fertility or Hungary or Poland for dental treatment. Yet, this did not appear the case for all medical tourism destinations, eg, while India clearly is a destination for medical tourists, this is for a whole broad spectrum of treatments.

Few studies were able to quantify patient flows and calculate effect on recipient health systems and the economy. Evidence does suggest that the inflow of medical travelers can increase inequities within the recipient country health systems 42 but that depends on the context. 11

Perhaps the most surprising finding was the increase in papers presenting primary research—a shortfall or gap that had been noted by the earlier literature reviews. 5,55,56 The recent publication date of many confirms the increase in research of medical travel.

Implications for the UK NHS

Evidence demonstrates that UK patients travel abroad to receive treatment and return with complications or infections that require follow‐up in the public sector. Based on papers reviewed, cosmetic procedures appear an area of growth for medical travel by UK patients and likely to result in cost to the NHS due to resulting complications, but costs resulting from other types of travel, including fertility and dental tourism, are evident. While complications experienced by UK medical tourists were reported, these were not compared to rates of complications for similar procedures undertaken in the UK, which would have further strengthened such research. However, in individual cases of patients described, these often focused on cases so unusual or extreme that the comparison or lack thereof to the UK was implicit. Case studies also underlined the challenges relating to information and communication, with often limited patient records available for returning medical tourists.

Despite a number of studies focusing on UK patients, overall the evidence presented underlines the need for further research to ascertain the potential impact and costs arising from medical tourism on the NHS. Only one study 42 estimated actual costs arising from complications of returning medical tourists and this was based on a small sample of patients. We found no research calculating the potential savings arising from UK patients traveling abroad for treatment. While research on risks associated with medical travel proved limited, the documented NDM1 outbreak in the UK highlighted the potential of infections that may result from medical travel. 36

Research focused on communication materials and websites highlights the lack of credible information about qualification and an absence of regulation and legal safeguards. This lack of clear information paired with the increasing willingness to travel of the UK public makes a greater numbers of complications a likely scenario.

Considering findings from the literature focused on the UK, these are particularly salient for the NHS at a time of reform. The lifting of the cap on private patients increases the potential for greater earning and marketing of NHS hospitals to foreign private patients. In this context, the lack of evidence on incoming tourists limits the possibility of informed decision making. Moreover, findings about complications of returning medical tourists, which highlight the need of quality control and continuity of care, are likely to mirror some of the policy challenges that will become evident in the implementation of the EU Directive on cross‐border health care implemented from 2013. In this context, it seems opportune for policymakers within the EU to further explore lessons from medical travel.

This review of the literature highlights a growing trend in medical travel that is likely to continue and have an increasing impact on patients, and originating and recipient health systems. It shows a diverse industry and different types of tourism depending on treatment, each with a complex set of patient motivation. Evidence also highlights complications experienced by patients, resulting in health problems and costs to originating health systems. While the review shows an increase in research over the past 2 years, it also clearly identifies limits to current knowledge and areas where the need for further research is evident:

A lack of information about patients' background and numbers of patients traveling abroad for treatment persists. The lack of data also restricts analysis about possible cost and benefits of medical travel.

Limited insights on why some patients travel when others do not.

Little is known about the industry beyond reviews of information materials and websites. Further research is needed to better understand how the sector operates, to ultimately understand impact on health services and outcomes of medical travelers.

Moreover, there is an absence of research examining the long‐term health outcomes of medical tourists when compared to patients treated within their country of residence. As a result, evidence on the comparative effect of treatment received abroad is lacking. Further qualitative and quantitative research beyond immediate clinical outcomes is needed to truly understand the effect of medical travel on patients, and its cost to the health system.

As medical tourism is set to rise, addressing these gaps in the evidence is urgently required to avoid potential harm to patients and health systems by enabling more informed policymaking on aspects of medical tourism.

This is a hospital in Hualien, Taiwan, which has two prominent advertising boards on its front facade promoting the hospital. Taiwan is a hot spot for medical tourism in Asia, attracting thousands of medical tourists mostly from China. Photo Credit: Eric Caumes .

The authors wish to acknowledge the members of the study advisory committee for their input into the review. This project was funded by the National Institute for Health Research Health Services and Delivery Research Programme (project number HSR 09/2001/21). The views and opinions expressed herein are those of the authors and do not necessarily reflect those of the HS&DR Programme, NIHR, NHS, or the Department of Health.

The authors state that they have no conflicts of interest to declare.

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Wellbeing and Resilience in Tourism: A Systematic Literature Review During COVID-19

Margarida pocinho.

1 CIERL, University of Madeira, Funchal, Portugal

2 Research Centre for Tourism, Sustainability and Well-Being (CinTurs), University of Algarve, Faro, Portugal

Soraia Garcês

Saúl neves de jesus, associated data.

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

The United Nations World Tourism Organization (UWTO) has acknowledged 2020 as the worst year in tourism history due to the worldwide pandemic COVID-19. Destinations, tourists, local communities, stakeholders, and residents, and their daily activities were affected. Thus, wellbeing and resilience are two crucial variables to help the industry and the people recover. This research aims to analyze early positive approaches and attitudes to respond to the negative impact of COVID-19 in tourism everyday activities that have at its core wellbeing and resilience, the two main variables of the Positive Psychology field of studies. A systematic literature review was conducted, following PRISMA guidelines to achieve this aim. The research was done using the Online Knowledge Library (B-on) and all the available databases. The research led to 32 articles that were screened using the inclusion and exclusion criteria. A total of 18 scientific articles met all criteria. Overall, results show that a positive and resilient approach to deal with the adverse outcomes of the pandemic is a concern for stakeholders and the future of the organizations in the tourism and hospitality sector, as is tourists’ wellbeing. However, less research has been done on wellbeing and a clear lack of research regarding residents’ wellbeing and resilience is evident. A deeper study of wellbeing and resilience in tourism is needed, and actual practices and interventions to ensure that all tourism actors have the resources to overcome the pandemic and restart the industry’s daily lives feeling well and safe.

Introduction

In 2015, the United Nations launched the 2030 agenda for Sustainable Development with 17 goals to transform the world. The overall aim of this agenda was (and it still is) to promote a more peaceful, resilient, and equitable world while keeping in mind the sustainability of the planet ( The Lancet Public Health, 2020 ). The array of Sustainable Development Goals (SDGs) includes health and wellbeing as one of these and assumes it as a priority for all ages ( The Lancet Public Health, 2020 ).

Wellbeing can be seen as a practice or a process related to living a good life ( Buzinde, 2020 ). The study of wellbeing has in Positive Psychology one of its main streams since this field is “(…) the scientific study of the strengths, characteristics, and actions that enable individuals and communities to thrive” ( Seligman, 2013 , p. 2).

Tourism can be a direct or indirect contributor to all sustainable goals ( Santos et al., 2020 ), including wellbeing. Scholars have considered Positive Psychology and subsequently, the study of wellbeing in tourism a natural step in the field that can support product innovation, the tourism experience, and leads to the competitiveness of tourism ( Garcês et al., 2020 ). Tourism has three base and important actors: tourists, destinations/locals, and stakeholders/workers. A balance between these is crucial to ensure the continuous improvement of the industry because one cannot exist without the other ( Garcês et al., 2020 ). Tourism experiences can improve the wellbeing of residents and tourists, and wellbeing can be a creative opportunity to innovate in destinations ( Garcês et al., 2018 ). However, studies in this field have focused mainly on tourists, with a noticeable lack of research about positive psychology variables focused on local communities and tourism workers ( Vada et al., 2020 ). Nevertheless, research has shown that tourists’ wellbeing is influenced by relationships, learning of a new place and culture, and/or learning new skills. Thus, initiatives that involve tourists within the community, such as volunteer activities, will promote tourists’ wellbeing, but not only locals, the community and even the place sustainability can gain from these experiences ( Vada et al., 2020 ).

However, COVID-19 led tourism activities to an unprecedented loss worldwide. From January 2020 to March 2021, there were 180 million fewer arrivals worldwide ( UNWTO, 2021b ). The lowest numbers were seen in Asia and the Pacific, followed by Europe, Africa, Middle East, and the Americas ( UNWTO, 2021b ). In January 2021, the number of international tourists’ arrivals was 87% less than in January 2020 ( UNWTO, 2021c ). In February 2021, 32% of worldwide destinations were entirely shut down to international arrivals, 34% partly closed, and only 2% have relaxed travel restrictions ( UNWTO, 2021a ). From an economic perspective, the pandemic led to a drop of 64% in receipts ( UNWTO, 2021b ). Destinations, residents, and tourists were (and still are) affected by the travel restrictions. While hope for improvement exists, particularly with the vaccination, experts believe that achieving 2019 numbers will only be possible after 2024 and maybe later ( UNWTO, 2021b ).

The pandemic is a threat to progress made in the sustainable development goal tree that looks to ensure health and wellbeing for all ( The Lancet Public Health, 2020 ). It has had severe consequences in society, the environment, and in people’s health and wellbeing ( Passavanti et al., 2021 ). Pandemics and other health crises lead to a growth in mental health problems, influencing tourists’ behaviors, and also their wellbeing ( Abbas et al., 2021 ). However, COVID-19 will also impact the well-being of those who work in tourism ( McCartney et al., 2021 ). Unemployment, panic generated by COVID-19, and lack of social support are considered key hazards to the tourism and hospitality employees’ perceived wellbeing ( Chen, 2020 ). In already done studies, in the context of COVID-19, job insecurity has had a significant effect on hotel employees’ anxiety and depression, and resilience was a moderator reducing the negative impact of job insecurity in depression ( Aguiar-Quintana et al., 2021 ). Overall, research shows that COVID-19 pandemic–perceived risk produces uncertainty and fear, leading to increased stress and vulnerability, and subsequently to a loss of mental wellbeing ( Paredes et al., 2021 ). Threat severity and susceptibility can trigger fear of traveling, yet it can also lead to protective travel behaviors. The fear of traveling can induce coping strategies, increasing individuals’ resilience, and embracing careful travel behaviors ( Zheng et al., 2021 ).

With the ongoing pandemic tourists, behavior patterns are expected to change, with tourists preferably choosing destinations with a low number of tourists and good sanitary conditions. A preference for places with outdoor activities or nature-related are characteristics looked for, as well as domestic destinations within the residency country. International travel has in destinations with a low number of COVID cases an attraction factor also ( Santos et al., 2020 ).

Recovering from COVID-19 has become a tourism research urgency, and the importance of resilience is clear to help build a quick and effective response and is a significant part of the ongoing research ( McCartney et al., 2021 ). Resilience is a concept that moves around “(…) between disciplines, between academia and public use, or between contexts,” and “it takes on slightly different meanings as it moves” ( Rogers et al., 2020 , p. 4). Resilience can be seen as a capacity to resist being “put down,” but also as the ability to recover and thrive from traumatic situations ( Harms et al., 2018 ). As a personal trait, resilience may allow people to manage negative situations better ( Liu et al., 2020 ). Research is showing that resilience has a mediator effect between personality traits and subjective wellbeing and stress experienced at the start of the pandemic, which leads to considering resilience as a protective factor to an adaptive reaction in the face of stressful experiences ( Kocjan et al., 2021 ). Expanding this concept, “The resilience level of how the community responds to the disruption caused by the lockdown and stress caused could influence city resilience” ( McCartney et al., 2021 , p. 7). Research in resilience must go beyond how to come out of a crisis and develop future resilience ( McCartney et al., 2021 ). Tourism must increase its resilience. This can be made by diversification: develop new business models; improve sustainability and digitalization ( Santos et al., 2020 ). As scholars are acknowledging, “From now on, the bet should not be on the increase in visitor numbers but on ‘better, more comfortable travel, personalized service, while maintaining affordable prices”’ ( Abbas et al., 2021 , p. 6).

The changes in tourists’ behaviors with the pandemic allow an opportunity for research and innovation in tourism. A preference for safe and healthy environments is expected. Also, a shift from overtourism destinations to less busy ones, emphasizing rural and nature tourism, is already being seen. This can be an opportunity to help places that are more remote to flourish, and at the same time diminish the effects of overtourism in others, as seen before the pandemic ( Santos et al., 2020 ). But also, the changes in tourists’ preferences can be opportunities to achieve the Sustainable Development Goals ( Santos et al., 2020 ), including the goal for health and wellbeing. As acknowledged by scholars “(…) in wellbeing, it is possible to have multiple directions and starting points. It, however, makes sense that research should be venturing toward new grounds and eudaimonic wellbeing seems a natural approach as it is a concept related to the idea of personal fulfillment and development that people are looking for” ( Garcês et al., 2020 , p. 113).

Thus, for the current research wellbeing and resilience were chosen as the main variables. Wellbeing was chosen because, beyond being a European Sustainable Goal for 2030, it is also a rising motivator for traveling and tourism. Resilience was chosen because it is a concept intimately related to wellbeing, that helps to deal effectively with adversity. Beyond that, it is part of many countries’ strategic planning to deal with the pandemic. So, considering the importance of wellbeing for tourism and the need for resilience for all actors of this industry, this study tries to answer the question of how are wellbeing and resilience being used in tourism as positive strategies to deal with the pandemic negative consequences? Particularly, this research aims to analyze early positive approaches and attitudes to respond to COVID-19 negative impact on tourism everyday activities that have at its core wellbeing and resilience, two main variables of the Positive Psychology field of studies, which is the theoretical framework that guides this current research. Tourism everyday activities in the current study were considered people’s (tourists, residents, workers/stakeholders) actions and behaviors in activities linked to tourism.

A systematic literature review was conducted using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses – PRISMA ( Moher et al., 2009 ). Considering the study aim, the following search terms were chosen Wellbeing; Resilience; Tourism; and Pandemic. The research took place in January 2021 using the Online Knowledge Library (B-On) and all the available databases on this platform which include: Complementary Index, SCOPUS, Academic Search Complete; Science Citation Index, Business Source Complete; MEDLINE, Supplemental Index, ScienceDirect; Directory of Open Access Journals, Social Sciences Citation Index, IEEE Xplore Digital Library, arXiv, Gale in Context: Science; Library, Information Science & Technology Abstracts, Arts and Humanities Citation Index, Gale Literature Resource Center, ERIC, SciELO, SciTech Connect, RCAAP, Dialnet, Government Printing Office Catalog, University Press Scholarship Online, Research Starters, Digital Access to Scholarship at Harvard (DASH), UC Digitalis; Oxford Scholarship online; SSOAR – Social Science Open Access Repository; eBook Index, Oxford Handbooks Online; and OAPEN Library. The search focused on scientific articles published between 2020 and 2021 in the English language.

The inclusion criteria used were (a) scientific articles published between 2020 and 2021; (b) articles written in English; (c) articles with the search terms included in its keywords; (d) scientific research articles with peer review; and (e) articles mainly focused on the search terms. The exclusion criteria used were (a) scientific articles published before 2020; (b) articles not written in English; (c) articles that did not include the search terms; (d) articles not peer-reviewed; and (e) articles not mainly focused on the search terms. The search was focused on the article’s keywords since this represents the core concepts of the articles.

The first search done on B-On crossed “Wellbeing or wellbeing or well-being” AND “Tour?sm*” AND “Pandemic or COVID-19 or coronavirus.” The second search was also done on B-On crossed the search terms: “Resilient?e” AND “Tour?sm*” AND “Pandemic or COVID-19 or coronavirus.” The Boolean operator “AND” was used to ensure that all three terms were included in the search and “OR” to ensure all variations for the terms “wellbeing” and those related to the “Pandemic.” The truncation symbol “*” was used to guarantee the inclusion of words with the same origin, and the “?” to include singular and plural forms. Inclusion/exclusion criteria (a); (b), (c), and (d) were applied through the online features of B-On, and criteria (e) was done manually.

Research led to the identification of 32 records in the following databases: Directory of Open Access Journals, Social Sciences Citation Index, ScienceDirect, Supplemental Index, Complementary Index, and SCOPUS. Four duplicates were found and removed, leading to 28 articles. Further analysis led to the exclusion of 11 more articles with criteria violations, namely, six commentaries; one editorial; and four records not mainly focused on the search terms. These criteria violations were encountered after applying inclusion/exclusion criteria (e) through a qualitative screening of each article abstract and/or full-text. Thus, in the end, a total of 17 scientific articles were considered as meeting all inclusion criteria, and hence, were further analyzed.

From the systematic literature review, only 32 records were first found. The application of the inclusion and exclusion criteria led to a total of 17 studies to be included in the final sample. The PRISMA ( Moher et al., 2009 ) flow diagram for this research can be seen in Figure 1 .

Flow diagram following PRISMA (2009) guidelines.

Despite the small number of articles, it is an indicator of interest by researchers on the importance of thinking about wellbeing and resilience amid the COVID-19 pandemic. Despite having restricted the search to 2020 and 2021, this decision was made solemnly with the intend to analyze the most current research regarding the use of positive variables such as wellbeing and resilience during the pandemic, which was only acknowledged by the World Health Organization (WHO) as a pandemic in March 2020 ( World Health Organization [WHO], 2020 ). Table 1 presents the number of articles published in 2020 and January 2021 (data collection retrieval month).

Number of articles distributed between 2020 and 2021.

From Table 1 , it is possible to acknowledge a low number of published articles related to the search terms on the selected dates. However, despite the low number of published articles, this research was done at the end of January 2021, almost a year after COVID-19 has been declared a pandemic ( World Health Organization [WHO], 2020 ). In this short time gap, the existence of already published materials at this time highlights the current need to learn and explore more the impact of the pandemic and how to restart tourism’s everyday activities with a positive outlook.

From the analysis of Table 2 , a clear emphasis is made on the resilience concept, with three times more articles on this topic than on wellbeing. This leads to thinking resilience as a positive psychological construct that reflects the skills to deal with adversity and is seen as important and as a positive asset and attitude to ensure the survival and future thriving of tourism.

Number of articles distributed according to the positive variables: Wellbeing and resilience.

In Table 3 , all assessed articles’ main findings are summarized.

Articles’ main findings.

From an in-depth analysis of the sample findings, it is possible to see two major tourism actors of interest: tourists and businesses. Regarding tourists, there is a wide range of topics studied. Some examined tourists’ perceptions about wellbeing, highlighting the importance of safety measures before travel or even upon arrival, such as getting tested for COVID-19 ( Agrusa et al., 2021 ). Others ( Yang et al., 2020 ) emphasized the need to revisit travel-induced wellbeing, and the need to rethink it, particularly in the long term, since some tourism experiences such as the ones that occurred during the COVID-19 pandemic, brought a loss of wellbeing after the trip, particularly perceived discrimination, thus questioning the literature that considers tourism as an induce-wellbeing activity ( Yang et al., 2020 ). Wellbeing was also considered as an important variable in predicting attitudes toward international travel and temporal avoidance behaviors ( Chua et al., 2020 ). Another research ( Zheng et al., 2021 ) studying different psychological variables and travel behavior in Chinese tourists, highlighted the positive benefits of “(…) psychological resilience on individuals’ intention to adapt caution travel after the pandemic outbreak.” Other researchers ( Wen et al., 2020 ) predicting how COVID-19 will affect tourists’ behaviors emphasized a growing interest in health and wellness tourism, among others. Another one of the studies ( Buckley and Westway, 2020 ) also acknowledged the psychological positive effects of walking-in-nature tourism, emphasizing an entrepreneur opportunity to promote wellbeing, particularly when thinking about recovering from the lockdowns that COVID-19 brought to the world.

As highlighted before, businesses were a focus of interest that stand-out in the current systematic research. In this regard, the importance of resilience to sustainable tourism development amid the pandemic is clear ( Sobaih et al., 2021 ). Also, the importance of organizational learning and business preparedness to deal with crisis and disasters is emphasized, which can lead to business resilience to overcome the negative impacts of such disasters, not only COVID-19, but future ones too ( Bhaskara and Filimonau, 2021 ). The positive impact of resilience on business performance is also highlighted in another study findings ( Setthachotsombut and Sua-iam, 2020 ). The need for businesses to implement actions and changes to cope with the pandemic and its impact is clear ( Alonso et al., 2020 ). Some studies in this regard highlight that satisfied employees with the organization COVID-19 responses positively influence job performance. Employees’ satisfaction may help to maintain their wellbeing, and therefore, they reciprocate through positive behaviors/attitudes ( Vo-Thanh et al., 2020 ). Again, the importance of corporate social responsibility to maintain not only employee’s resilience but also other positive psychological variables such as self-efficacy, hope, and optimism is empathized ( Mao et al., 2020 ). The importance of organizational resilience to organizational commitment, and the fact that resilience influences “the scope of adoption of anti-COVID-19 measures” is again emphasized ( Filimonau et al., 2020 ). Overall, another research stated that different sectors of the leisure and hospitality industry showed different resilience “levels” and some signs of recovery, still the pandemic is a hard situation and will endure a long-run recovery period ( Khan et al., 2020 ).

While tourists and businesses have a clear interest in research, some studies also highlight tourism as a whole unit, acknowledging the importance of resilience of destinations, enterprises, and tourists and its study, but also to see COVID-19 as an opportunity to reset tourism ( Prayag, 2020 ). A resilience framework for the tourism industry, highlighting this positive variable importance is acknowledged, and that smaller enterprises can also gain and ensure wellbeing at a bigger scale, while also promoting a more sustainable tourism ( Sharma et al., 2021 ). Research about COVID-19 can be a way to innovate tourism having sustainability and wellbeing as centerpieces ( Sigala, 2020 ).

In this analysis, it is also an important topic to rethink the future of tourism in the post-COVID era ( Agrusa et al., 2021 ). COVID-19 can be an opportunity to rethink tourism policies and strategies to ensure stability between the wellbeing of residents, tourists, and products, particularly in areas where overtourism was already a big issue among residents ( Agrusa et al., 2021 ).

Overall, the current systematic literature review highlighted the importance of wellbeing and resilience in tourism’s everyday activities during the COVID-19 pandemic. Tourism is one of the industries worldwide, that was most affected (if not the most affected) by the pandemic with a reduction of international arrivals from January 2020 to 2021 of more than 80% ( UNWTO, 2021c ). This number is astonishing and something that has never occurred before such a scale.

The pandemic is a threat to most activities in tourism and in many other sectors, and for all Humankind. Particularly it puts at risk the achievement of 2030 Sustainable Development Goals, including the mental health and wellbeing of all people ( Passavanti et al., 2021 ). Tourism was (and still is) tremendously affected by COVID-19, with a drop of more than 64% in receipts ( UNWTO, 2021b ), thus, affecting destinations, locals, and tourists. Resilience has come as a major goal and key process to overcome the challenges imposed by the pandemic. A big emphasis of this concept is seen on tourism stakeholders who are trying to survive COVID-19 impacts ( McCartney et al., 2021 ). The urgency to promote and increase resilience can be seen in the number of articles that focused on resilience comparatively to wellbeing in the current systematic literature review. The uncertainty that the pandemic brought made it urgent for the tourism industry to find new ways to overcome the difficulties. This can be observed in the results, where topics related to how to deal with the current crisis and even future ones ( Alonso et al., 2020 ; Bhaskara and Filimonau, 2021 ) or the benefits of resilience for businesses ( Setthachotsombut and Sua-iam, 2020 ) are highlighted and accentuated by the research. Resilience plans should be standard practice for all tourism stakeholders. While a pandemic was not something foreseen, it should be thought of as a warning for better planning and management in case of disasters or crises in tourism. Being an economic activity, tourism, highly dependent on external factors, such as weather, social crisis, or security, going forward, destination’s policymakers must prevent future crisis setbacks by planning and ensuring resilience to deal with whatever the next threat may be. In practical terms, such planning should be considered in national and local policies, but also as an internal business policy. In here, the introduction of, for example, policies to work remotely can be identified to ensure a smooth transition from on-site to on-line or even hybrid performances.

Another important result from this systematic analysis is the fact that research in wellbeing and resilience has mainly been focused on tourists and above all on businesses. This situation is also acknowledged by the literature ( Vada et al., 2020 ) where research has mostly been centered on tourists leaving behind local communities and tourism workers. Businesses have a particular interest in how to face COVID-19 and be resilient to ensure the thriving of the industry. Although in this review tourism workers were acknowledged in some articles ( Mao et al., 2020 ; Vo-Thanh et al., 2020 ), more needs to be done because their wellbeing is also affected by the pandemic ( McCartney et al., 2021 ) and they are one of the key pillars of tourism ( Garcês et al., 2020 ). Thus, businesses should invest more in wellbeing of their workers since they are the forefront of tourism businesses but also its background. A worker that feels safe and secure will deliver a more satisfactory service that will in return improve the tourists’ satisfaction. However, the complete lack of research on local communities and their residents is of concern, because the pandemic has affected global livelihoods and destinations have no longer tourists ( Abbas et al., 2021 ), which will have impacts on places that have tourism as the main economy. Therefore, studies about locals’ wellbeing and how they face pandemic’s incoming repercussions should be developed, which will hopefully lead to the development of strategies to help the residents of tourism destinations deal with this crisis aftermath while promoting their wellbeing and mental health.

Another interesting result is the disparity between the articles that focused on wellbeing and resilience. Resilience shows a far greater interest. However, it is important to not forget that tourism can be a way to experience wellbeing ( Garcês et al., 2020 ), and although the number of articles focused on wellbeing is much less, they show that this variable should not be forgotten, highlighting the fact that COVID-19 will affect the interest of tourists, moving them to destinations that have in attention wellness and wellbeing endeavors ( Wen et al., 2020 ). Policymakers and stakeholders have in here a “gold” opportunity to innovate. Wellbeing and wellness can be attractive factors for new tourists and thus open doors to developing new products and activities in destinations. These changes in tourists’ behaviors should be seen as opportunities to “refresh” tourism and even to solve pre-COVID problems in some destinations such as overtourism. Tourists will be now looking more for quieter places, with outdoor experiences and in nature ( Santos et al., 2020 ). This is interesting because research in pre-COVID times has linked nature to feelings of wellbeing ( Garcês et al., 2018 ), and results in the current study already show this trend highlighting the psychological benefits of walking in nature, for example ( Buckley and Westway, 2020 ). Thus, stakeholders can have in wellness and nature products a source to innovate and promote tourists’ wellbeing.

It is also important to note that a small number of articles in this systematic review highlight the future of tourism after COVID-19. This emphasis gives hope for a positive outlook for the future, focusing on the proactive and preventive measures that will help the survival and thriving of the industry, highlighting the importance of developing new policies and strategies to promote wellbeing among all tourism actors ( Agrusa et al., 2021 ). This idea is in accordance with the literature that acknowledges the need to focus on the quality and personalization of services at reasonable prices ( Abbas et al., 2021 ), build new business models, and enhance sustainability and digitalization ( Santos et al., 2020 ) on the post-COVID era. But to become a more resilient industry and a promoter of wellbeing, the industry needs to first think of what has gone wrong, identify the stressors, and develop contingency plans and strategic ones to deal with present and future uncertainty, including crisis. Additionally, it is relevant to notice that the new emerging field of existential positive psychology focus the importance of suffering to flourishing and highlights that dealing with negative situations will allow its transformation to a sense of accomplishment and mature happiness ( Wong, 2021 ). In this new emerging trend, wellbeing is simultaneously a process and an outcome in dealing with the search for positive life goals and transforming the negative situations into better ones ( Wong, 2020 ). Thus, whilst the pandemic has had so far, a tremendous negative impact in tourism, it can also be seen as an opportunity to innovation, and to build the sector resilience while promoting wellbeing for the destinations, the stakeholders, and the tourists. With this new vision of existential Positive Psychology, it is possible to reflect that while COVID-19 brought with it a lot of suffering including for tourism stakeholders, it is also an opportunity to learn from and develop new strengths and simultaneously to improve people tourism experiences.

This study thus highlights an interest in resilience and wellbeing in tourism. However, there is still space for growth. Destinations’ policymakers and all involved in tourism must prepare better for a future crisis, with resilience programs that consider strategies to surpass the adverse outcomes of such crisis, not only for businesses but also for their workers and their residents. At the same time, developing innovation through wellbeing can be a differentiator factor for the destinations, but it can also help promote tourists, locals, and stakeholders’ mental health. New products with a focus on wellness, nature, or rural places can be starting points. This research also brings with it the importance of (re)thinking tourism not only in economic gains, but also in psychological aspects that can have great impact on the restart of the tourism during this pandemic and beyond. Also, this study was developed considering only the COVID-19 pandemic, and what has been done during this situation in terms of wellbeing and resilience in tourism. Thus, it shows the impact of COVID-19 pandemic, a current world problem, in tourism studies. Additionally, world institutions such as OMS and governments of many countries have highlighted the need to be resilient and promote wellbeing in the face of this health crisis, and the tourism industry, as one of the most affected, is no exception.

As for the study limitations, this research was done in January 2021, thus throughout the year, there is an expectation that more studies about the impact of COVID-19 in tourism, particularly considering the study variables will have been published. Thus, it will be important to further study these variables in future studies. The use of keywords may have limited the scope of the research. Therefore, in future research, expanding the search to the title and abstract may allow for the generation of a more significant number of articles on these topics. Also, it will be important to develop practical interventions on how wellbeing and resilience can be key points in tourism. Thus, not only tourists or businesses should be the focus, but also the locals and the workers of this sector.

Overall, in this systematic literature review, it was possible to see a clear focus on the impact and recovery of businesses from COVID-19 pandemic, with resilience as an important variable to achieve it. It is the authors’ belief that this article can contribute particularly to those countries and regions that exclusively depend on tourism everyday activities and that were severely affected by the pandemic, since many depend entirely on the tourism industry to economically survive. This article also hopes to contribute with some ideas and suggestions of how to introduce wellbeing and resilience in the tourism industry, allowing for potential course of action to be taken by all those involved in it. Concluding, while the articles that met all the inclusion criteria were few, this systematic review highlights the concerns of the sector and the urgency to rebound quickly and effectively, and restart tourism and its everyday activities safely and with a positive attitude.

Data Availability Statement

Author contributions.

All authors listed have made a substantial, direct, and intellectual contribution to the work, and approved it for publication.

Conflict of Interest

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

Publisher’s Note

All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.

This article was financed by the National Funds provided by the FCT - Foundation for Science and Technology through project UIDB/04020/2020.

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Health Tourism-Subject of Scientific Research: A Literature Review and Cluster Analysis

Affiliations.

• 1 Department of Tourism, Social Communication and Consulting, Institute of Economics and Finance, Warsaw University of Life Sciences, Nowoursynowska 166, 02-787 Warsaw, Poland.
• 2 Department of Logistics, Institute of Economics and Finance, Warsaw University of Life Sciences, Nowoursynowska 166, 02-787 Warsaw, Poland.
• 3 Faculty of Economics, Poznań University of Life Sciences, Wojska Polskiego 28, 60-637 Poznań, Poland.
• PMID: 36612800
• PMCID: PMC9819796
• DOI: 10.3390/ijerph20010480

The purpose of this article is to identify main research areas in health tourism in scientific research. The data used in this analysis span from 2000 to 2022, was retrieved from the Web of Science database, and comprises a total of 1493 bibliometric records of publications. The paper includes both a quantitative and a qualitative analysis. The following four main research areas were identified based on the results: (1) patient satisfaction built upon trust; (2) health impacts of the destination (including the economic aspect, which plays a decisive role in choosing a tourism destination); (3) health behavior as a major part of human activity; and (4) traveling with a view to regain one's health. Note that the limitations of this study-which mostly affect the methodological part-need to be taken into consideration. This is the consequence of the selected publication database and of the search criteria used, such as the publication year or language.

Keywords: cluster analysis; co-word; literature review; tourism; tourism economics; trend.

Publication types

• Bibliometrics
• Cluster Analysis
• Health Behavior
• Medical Tourism*

Grants and funding

• Open access
• Published: 15 May 2024

Learning together for better health using an evidence-based Learning Health System framework: a case study in stroke

• Helena Teede 1 , 2   na1 ,
• Dominique A. Cadilhac 3 , 4   na1 ,
• Tara Purvis 3 ,
• Monique F. Kilkenny 3 , 4 ,
• Bruce C.V. Campbell 4 , 5 , 6 ,
• Coralie English 7 ,
• Alison Johnson 2 ,
• Emily Callander 1 ,
• Rohan S. Grimley 8 , 9 ,
• Christopher Levi 10 ,
• Sandy Middleton 11 , 12 ,
• Kelvin Hill 13 &
• Joanne Enticott   ORCID: orcid.org/0000-0002-4480-5690 1  

BMC Medicine volume  22 , Article number:  198 ( 2024 ) Cite this article

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Metrics details

In the context of expanding digital health tools, the health system is ready for Learning Health System (LHS) models. These models, with proper governance and stakeholder engagement, enable the integration of digital infrastructure to provide feedback to all relevant parties including clinicians and consumers on performance against best practice standards, as well as fostering innovation and aligning healthcare with patient needs. The LHS literature primarily includes opinion or consensus-based frameworks and lacks validation or evidence of benefit. Our aim was to outline a rigorously codesigned, evidence-based LHS framework and present a national case study of an LHS-aligned national stroke program that has delivered clinical benefit.

Current core components of a LHS involve capturing evidence from communities and stakeholders (quadrant 1), integrating evidence from research findings (quadrant 2), leveraging evidence from data and practice (quadrant 3), and generating evidence from implementation (quadrant 4) for iterative system-level improvement. The Australian Stroke program was selected as the case study as it provides an exemplar of how an iterative LHS works in practice at a national level encompassing and integrating evidence from all four LHS quadrants. Using this case study, we demonstrate how to apply evidence-based processes to healthcare improvement and embed real-world research for optimising healthcare improvement. We emphasize the transition from research as an endpoint, to research as an enabler and a solution for impact in healthcare improvement.

Conclusions

The Australian Stroke program has nationally improved stroke care since 2007, showcasing the value of integrated LHS-aligned approaches for tangible impact on outcomes. This LHS case study is a practical example for other health conditions and settings to follow suit.

Peer Review reports

Internationally, health systems are facing a crisis, driven by an ageing population, increasing complexity, multi-morbidity, rapidly advancing health technology and rising costs that threaten sustainability and mandate transformation and improvement [ 1 , 2 ]. Although research has generated solutions to healthcare challenges, and the advent of big data and digital health holds great promise, entrenched siloes and poor integration of knowledge generation, knowledge implementation and healthcare delivery between stakeholders, curtails momentum towards, and consistent attainment of, evidence-and value-based care [ 3 ]. This is compounded by the short supply of research and innovation leadership within the healthcare sector, and poorly integrated and often inaccessible health data systems, which have crippled the potential to deliver on digital-driven innovation [ 4 ]. Current approaches to healthcare improvement are also often isolated with limited sustainability, scale-up and impact [ 5 ].

Evidence suggests that integration and partnership across academic and healthcare delivery stakeholders are key to progress, including those with lived experience and their families (referred to here as consumers and community), diverse disciplines (both research and clinical), policy makers and funders. Utilization of evidence from research and evidence from practice including data from routine care, supported by implementation research, are key to sustainably embedding improvement and optimising health care and outcomes. A strategy to achieve this integration is through the Learning Health System (LHS) (Fig.  1 ) [ 2 , 6 , 7 , 8 ]. Although there are numerous publications on LHS approaches [ 9 , 10 , 11 , 12 ], many focus on research perspectives and data, most do not demonstrate tangible healthcare improvement or better health outcomes. [ 6 ]

Monash Learning Health System: The Learn Together for Better Health Framework developed by Monash Partners and Monash University (from Enticott et al. 2021 [ 7 ]). Four evidence quadrants: Q1 (orange) is evidence from stakeholders; Q2 (green) is evidence from research; Q3 (light blue) is evidence from data; and, Q4 (dark blue) is evidence from implementation and healthcare improvement

In developed nations, it has been estimated that 60% of care provided aligns with the evidence base, 30% is low value and 10% is potentially harmful [ 13 ]. In some areas, clinical advances have been rapid and research and evidence have paved the way for dramatic improvement in outcomes, mandating rapid implementation of evidence into healthcare (e.g. polio and COVID-19 vaccines). However, healthcare improvement is challenging and slow [ 5 ]. Health systems are highly complex in their design, networks and interacting components, and change is difficult to enact, sustain and scale up. [ 3 ] New effective strategies are needed to meet community needs and deliver evidence-based and value-based care, which reorients care from serving the provider, services and system, towards serving community needs, based on evidence and quality. It goes beyond cost to encompass patient and provider experience, quality care and outcomes, efficiency and sustainability [ 2 , 6 ].

The costs of stroke care are expected to rise rapidly in the next decades, unless improvements in stroke care to reduce the disabling effects of strokes can be successfully developed and implemented [ 14 ]. Here, we briefly describe the Monash LHS framework (Fig.  1 ) [ 2 , 6 , 7 ] and outline an exemplar case in order to demonstrate how to apply evidence-based processes to healthcare improvement and embed real-world research for optimising healthcare. The Australian LHS exemplar in stroke care has driven nationwide improvement in stroke care since 2007.

An evidence-based Learning Health System framework

In Australia, members of this author group (HT, AJ, JE) have rigorously co-developed an evidence-based LHS framework, known simply as the Monash LHS [ 7 ]. The Monash LHS was designed to support sustainable, iterative and continuous robust benefit of improved clinical outcomes. It was created with national engagement in order to be applicable to Australian settings. Through this rigorous approach, core LHS principles and components have been established (Fig.  1 ). Evidence shows that people/workforce, culture, standards, governance and resources were all key to an effective LHS [ 2 , 6 ]. Culture is vital including trust, transparency, partnership and co-design. Key processes include legally compliant data sharing, linkage and governance, resources, and infrastructure [ 4 ]. The Monash LHS integrates disparate and often siloed stakeholders, infrastructure and expertise to ‘Learn Together for Better Health’ [ 7 ] (Fig.  1 ). This integrates (i) evidence from community and stakeholders including priority areas and outcomes; (ii) evidence from research and guidelines; (iii) evidence from practice (from data) with advanced analytics and benchmarking; and (iv) evidence from implementation science and health economics. Importantly, it starts with the problem and priorities of key stakeholders including the community, health professionals and services and creates an iterative learning system to address these. The following case study was chosen as it is an exemplar of how a Monash LHS-aligned national stroke program has delivered clinical benefit.

Australian Stroke Learning Health System

Internationally, the application of LHS approaches in stroke has resulted in improved stroke care and outcomes [ 12 ]. For example, in Canada a sustained decrease in 30-day in-hospital mortality has been found commensurate with an increase in resources to establish the multifactorial stroke system intervention for stroke treatment and prevention [ 15 ]. Arguably, with rapid advances in evidence and in the context of an ageing population with high cost and care burden and substantive impacts on quality of life, stroke is an area with a need for rapid research translation into evidence-based and value-based healthcare improvement. However, a recent systematic review found that the existing literature had few comprehensive examples of LHS adoption [ 12 ]. Although healthcare improvement systems and approaches were described, less is known about patient-clinician and stakeholder engagement, governance and culture, or embedding of data informatics into everyday practice to inform and drive improvement [ 12 ]. For example, in a recent review of quality improvement collaborations, it was found that although clinical processes in stroke care are improved, their short-term nature means there is uncertainty about sustainability and impacts on patient outcomes [ 16 ]. Table  1 provides the main features of the Australian Stroke LHS based on the four core domains and eight elements of the Learning Together for Better Health Framework described in Fig.  1 . The features are further expanded on in the following sections.

Evidence from stakeholders (LHS quadrant 1, Fig.  1 )

Engagement, partners and priorities.

Within the stroke field, there have been various support mechanisms to facilitate an LHS approach including partnership and broad stakeholder engagement that includes clinical networks and policy makers from different jurisdictions. Since 2008, the Australian Stroke Coalition has been co-led by the Stroke Foundation, a charitable consumer advocacy organisation, and Stroke Society of Australasia a professional society with membership covering academics and multidisciplinary clinician networks, that are collectively working to improve stroke care ( https://australianstrokecoalition.org.au/ ). Surveys, focus groups and workshops have been used for identifying priorities from stakeholders. Recent agreed priorities have been to improve stroke care and strengthen the voice for stroke care at a national ( https://strokefoundation.org.au/ ) and international level ( https://www.world-stroke.org/news-and-blog/news/world-stroke-organization-tackle-gaps-in-access-to-quality-stroke-care ), as well as reduce duplication amongst stakeholders. This activity is built on a foundation and culture of research and innovation embedded within the stroke ‘community of practice’. Consumers, as people with lived experience of stroke are important members of the Australian Stroke Coalition, as well as representatives from different clinical colleges. Consumers also provide critical input to a range of LHS activities via the Stroke Foundation Consumer Council, Stroke Living Guidelines committees, and the Australian Stroke Clinical Registry (AuSCR) Steering Committee (described below).

Evidence from research (LHS quadrant 2, Fig.  1 )

Advancement of the evidence for stroke interventions and synthesis into clinical guidelines.

To implement best practice, it is crucial to distil the large volume of scientific and trial literature into actionable recommendations for clinicians to use in practice [ 24 ]. The first Australian clinical guidelines for acute stroke were produced in 2003 following the increasing evidence emerging for prevention interventions (e.g. carotid endarterectomy, blood pressure lowering), acute medical treatments (intravenous thrombolysis, aspirin within 48 h of ischemic stroke), and optimised hospital management (care in dedicated stroke units by a specialised and coordinated multidisciplinary team) [ 25 ]. Importantly, a number of the innovations were developed, researched and proven effective by key opinion leaders embedded in the Australian stroke care community. In 2005, the clinical guidelines for Stroke Rehabilitation and Recovery [ 26 ] were produced, with subsequent merged guidelines periodically updated. However, the traditional process of periodic guideline updates is challenging for end users when new research can render recommendations redundant and this lack of currency erodes stakeholder trust [ 27 ]. In response to this challenge the Stroke Foundation and Cochrane Australia entered a pioneering project to produce the first electronic ‘living’ guidelines globally [ 20 ]. Major shifts in the evidence for reperfusion therapies (e.g. extended time-window intravenous thrombolysis and endovascular clot retrieval), among other advances, were able to be converted into new recommendations, approved by the Australian National Health and Medical Research Council within a few months of publication. Feedback on this process confirmed the increased use and trust in the guidelines by clinicians. The process informed other living guidelines programs, including the successful COVID-19 clinical guidelines [ 28 ].

However, best practice clinical guideline recommendations are necessary but insufficient for healthcare improvement and nesting these within an LHS with stakeholder partnership, enables implementation via a range of proven methods, including audit and feedback strategies [ 29 ].

Evidence from data and practice (LHS quadrant 3, Fig.  1 )

Data systems and benchmarking : revealing the disparities in care between health services. A national system for standardized stroke data collection was established as the National Stroke Audit program in 2007 by the Stroke Foundation [ 30 ] following various state-level programs (e.g. New South Wales Audit) [ 31 ] to identify evidence-practice gaps and prioritise improvement efforts to increase access to stroke units and other acute treatments [ 32 ]. The Audit program alternates each year between acute (commencing in 2007) and rehabilitation in-patient services (commencing in 2008). The Audit program provides a ‘deep dive’ on the majority of recommendations in the clinical guidelines whereby participating hospitals provide audits of up to 40 consecutive patient medical records and respond to a survey about organizational resources to manage stroke. In 2009, the AuSCR was established to provide information on patients managed in acute hospitals based on a small subset of quality processes of care linked to benchmarked reports of performance (Fig.  2 ) [ 33 ]. In this way, the continuous collection of high-priority processes of stroke care could be regularly collected and reviewed to guide improvement to care [ 34 ]. Plus clinical quality registry programs within Australia have shown a meaningful return on investment attributed to enhanced survival, improvements in quality of life and avoided costs of treatment or hospital stay [ 35 ].

Example performance report from the Australian Stroke Clinical Registry: average door-to-needle time in providing intravenous thrombolysis by different hospitals in 2021 [ 36 ]. Each bar in the figure represents a single hospital

The Australian Stroke Coalition endorsed the creation of an integrated technological solution for collecting data through a single portal for multiple programs in 2013. In 2015, the Stroke Foundation, AuSCR consortium, and other relevant groups cooperated to design an integrated data management platform (the Australian Stroke Data Tool) to reduce duplication of effort for hospital staff in the collection of overlapping variables in the same patients [ 19 ]. Importantly, a national data dictionary then provided the common data definitions to facilitate standardized data capture. Another important feature of AuSCR is the collection of patient-reported outcome surveys between 90 and 180 days after stroke, and annual linkage with national death records to ascertain survival status [ 33 ]. To support a LHS approach, hospitals that participate in AuSCR have access to a range of real-time performance reports. In efforts to minimize the burden of data collection in the AuSCR, interoperability approaches to import data directly from hospital or state-level managed stroke databases have been established (Fig.  3 ); however, the application has been variable and 41% of hospitals still manually enter all their data.

Current status of automated data importing solutions in the Australian Stroke Clinical Registry, 2022, with ‘ n ’ representing the number of hospitals. AuSCR, Australian Stroke Clinical Registry; AuSDaT, Australian Stroke Data Tool; API, Application Programming Interface; ICD, International Classification of Diseases; RedCAP, Research Electronic Data Capture; eMR, electronic medical records

For acute stroke care, the Australian Commission on Quality and Safety in Health Care facilitated the co-design (clinicians, academics, consumers) and publication of the national Acute Stroke Clinical Care Standard in 2015 [ 17 ], and subsequent review [ 18 ]. The indicator set for the Acute Stroke Standard then informed the expansion of the minimum dataset for AuSCR so that hospitals could routinely track their performance. The national Audit program enabled hospitals not involved in the AuSCR to assess their performance every two years against the Acute Stroke Standard. Complementing these efforts, the Stroke Foundation, working with the sector, developed the Acute and Rehabilitation Stroke Services Frameworks to outline the principles, essential elements, models of care and staffing recommendations for stroke services ( https://informme.org.au/guidelines/national-stroke-services-frameworks ). The Frameworks are intended to guide where stroke services should be developed, and monitor their uptake with the organizational survey component of the Audit program.

Evidence from implementation and healthcare improvement (LHS quadrant 4, Fig.  1 )

Research to better utilize and augment data from registries through linkage [ 37 , 38 , 39 , 40 ] and to ensure presentation of hospital or service level data are understood by clinicians has ensured advancement in the field for the Australian Stroke LHS [ 41 ]. Importantly, greater insights into whole patient journeys, before and after a stroke, can now enable exploration of value-based care. The LHS and stroke data platform have enabled focused and time-limited projects to create a better understanding of the quality of care in acute or rehabilitation settings [ 22 , 42 , 43 ]. Within stroke, all the elements of an LHS culminate into the ready availability of benchmarked performance data and support for implementation of strategies to address gaps in care.

Implementation research to grow the evidence base for effective improvement interventions has also been a key pillar in the Australian context. These include multi-component implementation interventions to achieve behaviour change for particular aspects of stroke care, [ 22 , 23 , 44 , 45 ] and real-world approaches to augmenting access to hyperacute interventions in stroke through the use of technology and telehealth [ 46 , 47 , 48 , 49 ]. The evidence from these studies feeds into the living guidelines program and the data collection systems, such as the Audit program or AuSCR, which are then amended to ensure data aligns to recommended care. For example, the use of ‘hyperacute aspirin within the first 48 h of ischemic stroke’ was modified to be ‘hyperacute antiplatelet…’ to incorporate new evidence that other medications or combinations are appropriate to use. Additionally, new datasets have been developed to align with evidence such as the Fever, Sugar, and Swallow variables [ 42 ]. Evidence on improvements in access to best practice care from the acute Audit program [ 50 ] and AuSCR is emerging [ 36 ]. For example, between 2007 and 2017, the odds of receiving intravenous thrombolysis after ischemic stroke increased by 16% 9OR 1.06 95% CI 1.13–1.18) and being managed in a stroke unit by 18% (OR 1.18 95% CI 1.17–1.20). Over this period, the median length of hospital stay for all patients decreased from 6.3 days in 2007 to 5.0 days in 2017 [ 51 ]. When considering the number of additional patients who would receive treatment in 2017 in comparison to 2007 it was estimated that without this additional treatment, over 17,000 healthy years of life would be lost in 2017 (17,786 disability-adjusted life years) [ 51 ]. There is evidence on the cost-effectiveness of different system-focussed strategies to augment treatment access for acute ischemic stroke (e.g. Victorian Stroke Telemedicine program [ 52 ] and Melbourne Mobile Stroke Unit ambulance [ 53 ]). Reciprocally, evidence from the national Rehabilitation Audit, where the LHS approach has been less complete or embedded, has shown fewer areas of healthcare improvement over time [ 51 , 54 ].

Within the field of stroke in Australia, there is indirect evidence that the collective efforts that align to establishing the components of a LHS have had an impact. Overall, the age-standardised rate of stroke events has reduced by 27% between 2001 and 2020, from 169 to 124 events per 100,000 population. Substantial declines in mortality rates have been reported since 1980. Commensurate with national clinical guidelines being updated in 2007 and the first National Stroke Audit being undertaken in 2007, the mortality rates for men (37.4 deaths per 100,000) and women (36.1 deaths per 100,0000 has declined to 23.8 and 23.9 per 100,000, respectively in 2021 [ 55 ].

Underpinning the LHS with the integration of the four quadrants of evidence from stakeholders, research and guidelines, practice and implementation, and core LHS principles have been addressed. Leadership and governance have been important, and programs have been established to augment workforce training and capacity building in best practice professional development. Medical practitioners are able to undertake courses and mentoring through the Australasian Stroke Academy ( http://www.strokeacademy.com.au/ ) while nurses (and other health professionals) can access teaching modules in stroke care from the Acute Stroke Nurses Education Network ( https://asnen.org/ ). The Association of Neurovascular Clinicians offers distance-accessible education and certification to develop stroke expertise for interdisciplinary professionals, including advanced stroke co-ordinator certification ( www.anvc.org ). Consumer initiative interventions are also used in the design of the AuSCR Public Summary Annual reports (available at https://auscr.com.au/about/annual-reports/ ) and consumer-related resources related to the Living Guidelines ( https://enableme.org.au/resources ).

The important success factors and lessons from stroke as a national exemplar LHS in Australia include leadership, culture, workforce and resources integrated with (1) established and broad partnerships across the academic-clinical sector divide and stakeholder engagement; (2) the living guidelines program; (3) national data infrastructure, including a national data dictionary that provides the common data framework to support standardized data capture; (4) various implementation strategies including benchmarking and feedback as well as engagement strategies targeting different levels of the health system; and (5) implementation and improvement research to advance stroke systems of care and reduce unwarranted variation in practice (Fig.  1 ). Priority opportunities now include the advancement of interoperability with electronic medical records as an area all clinical quality registry’s programs needs to be addressed, as well as providing more dynamic and interactive data dashboards tailored to the need of clinicians and health service executives.

There is a clear mandate to optimise healthcare improvement with big data offering major opportunities for change. However, we have lacked the approaches to capture evidence from the community and stakeholders, to integrate evidence from research, to capture and leverage data or evidence from practice and to generate and build on evidence from implementation using iterative system-level improvement. The LHS provides this opportunity and is shown to deliver impact. Here, we have outlined the process applied to generate an evidence-based LHS and provide a leading exemplar in stroke care. This highlights the value of moving from single-focus isolated approaches/initiatives to healthcare improvement and the benefit of integration to deliver demonstrable outcomes for our funders and key stakeholders — our community. This work provides insight into strategies that can both apply evidence-based processes to healthcare improvement as well as implementing evidence-based practices into care, moving beyond research as an endpoint, to research as an enabler, underpinning delivery of better healthcare.

Availability of data and materials

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Abbreviations

Australian Stroke Clinical Registry

Confidence interval

• Learning Health System

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Acknowledgements

The following authors hold National Health and Medical Research Council Research Fellowships: HT (#2009326), DAC (#1154273), SM (#1196352), MFK Future Leader Research Fellowship (National Heart Foundation #105737). The Funders of this work did not have any direct role in the design of the study, its execution, analyses, interpretation of the data, or decision to submit results for publication.

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Helena Teede and Dominique A. Cadilhac contributed equally.

Authors and Affiliations

Monash Centre for Health Research and Implementation, 43-51 Kanooka Grove, Clayton, VIC, Australia

Helena Teede, Emily Callander & Joanne Enticott

Monash Partners Academic Health Science Centre, 43-51 Kanooka Grove, Clayton, VIC, Australia

Helena Teede & Alison Johnson

Stroke and Ageing Research, Department of Medicine, School of Clinical Sciences at Monash Health, Monash University, Level 2 Monash University Research, Victorian Heart Hospital, 631 Blackburn Rd, Clayton, VIC, Australia

Dominique A. Cadilhac, Tara Purvis & Monique F. Kilkenny

Stroke Theme, The Florey Institute of Neuroscience and Mental Health, University of Melbourne, Heidelberg, VIC, Australia

Dominique A. Cadilhac, Monique F. Kilkenny & Bruce C.V. Campbell

Department of Neurology, Melbourne Brain Centre, Royal Melbourne Hospital, Parkville, VIC, Australia

Bruce C.V. Campbell

Department of Medicine, Faculty of Medicine, Dentistry and Health Sciences, University of Melbourne, Victoria, Australia

School of Health Sciences, Heart and Stroke Program, University of Newcastle, Hunter Medical Research Institute, University Drive, Callaghan, NSW, Australia

Coralie English

School of Medicine and Dentistry, Griffith University, Birtinya, QLD, Australia

Rohan S. Grimley

Clinical Excellence Division, Queensland Health, Brisbane, Australia

John Hunter Hospital, Hunter New England Local Health District and University of Newcastle, Sydney, NSW, Australia

Christopher Levi

School of Nursing, Midwifery and Paramedicine, Australian Catholic University, Sydney, NSW, Australia

Sandy Middleton

Nursing Research Institute, St Vincent’s Health Network Sydney and and Australian Catholic University, Sydney, NSW, Australia

Stroke Foundation, Level 7, 461 Bourke St, Melbourne, VIC, Australia

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HT: conception, design and initial draft, developed the theoretical formalism for learning health system framework, approved the submitted version. DAC: conception, design and initial draft, provided essential literature and case study examples, approved the submitted version. TP: revised the manuscript critically for important intellectual content, approved the submitted version. MFK: revised the manuscript critically for important intellectual content, provided essential literature and case study examples, approved the submitted version. BC: revised the manuscript critically for important intellectual content, provided essential literature and case study examples, approved the submitted version. CE: revised the manuscript critically for important intellectual content, provided essential literature and case study examples, approved the submitted version. AJ: conception, design and initial draft, developed the theoretical formalism for learning health system framework, approved the submitted version. EC: revised the manuscript critically for important intellectual content, approved the submitted version. RSG: revised the manuscript critically for important intellectual content, provided essential literature and case study examples, approved the submitted version. CL: revised the manuscript critically for important intellectual content, provided essential literature and case study examples, approved the submitted version. SM: revised the manuscript critically for important intellectual content, provided essential literature and case study examples, approved the submitted version. KH: revised the manuscript critically for important intellectual content, provided essential literature and case study examples, approved the submitted version. JE: conception, design and initial draft, developed the theoretical formalism for learning health system framework, approved the submitted version. All authors read and approved the final manuscript.

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Sexual and reproductive health implementation research in humanitarian contexts: a scoping review

• Alexandra Norton 1 &
• Hannah Tappis 2  

Reproductive Health volume  21 , Article number:  64 ( 2024 ) Cite this article

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Meeting the health needs of crisis-affected populations is a growing challenge, with 339 million people globally in need of humanitarian assistance in 2023. Given one in four people living in humanitarian contexts are women and girls of reproductive age, sexual and reproductive health care is considered as essential health service and minimum standard for humanitarian response. Despite growing calls for increased investment in implementation research in humanitarian settings, guidance on appropriate methods and analytical frameworks is limited.

A scoping review was conducted to examine the extent to which implementation research frameworks have been used to evaluate sexual and reproductive health interventions in humanitarian settings. Peer-reviewed papers published from 2013 to 2022 were identified through relevant systematic reviews and a literature search of Pubmed, Embase, PsycInfo, CINAHL and Global Health databases. Papers that presented primary quantitative or qualitative data pertaining to a sexual and reproductive health intervention in a humanitarian setting were included.

Seven thousand thirty-six unique records were screened for inclusion, and 69 papers met inclusion criteria. Of these, six papers explicitly described the use of an implementation research framework, three citing use of the Consolidated Framework for Implementation Research. Three additional papers referenced other types of frameworks used in their evaluation. Factors cited across all included studies as helping the intervention in their presence or hindering in their absence were synthesized into the following Consolidated Framework for Implementation Research domains: Characteristics of Systems, Outer Setting, Inner Setting, Characteristics of Individuals, Intervention Characteristics, and Process.

This review found a wide range of methodologies and only six of 69 studies using an implementation research framework, highlighting an opportunity for standardization to better inform the evidence for and delivery of sexual and reproductive health interventions in humanitarian settings. Increased use of implementation research frameworks such as a modified Consolidated Framework for Implementation Research could work toward both expanding the evidence base and increasing standardization.

Plain English summary

Three hundred thirty-nine million people globally were in need of humanitarian assistance in 2023, and meeting the health needs of crisis-affected populations is a growing challenge. One in four people living in humanitarian contexts are women and girls of reproductive age, and provision of sexual and reproductive health care is considered to be essential within a humanitarian response. Implementation research can help to better understand how real-world contexts affect health improvement efforts. Despite growing calls for increased investment in implementation research in humanitarian settings, guidance on how best to do so is limited. This scoping review was conducted to examine the extent to which implementation research frameworks have been used to evaluate sexual and reproductive health interventions in humanitarian settings. Of 69 papers that met inclusion criteria for the review, six of them explicitly described the use of an implementation research framework. Three used the Consolidated Framework for Implementation Research, a theory-based framework that can guide implementation research. Three additional papers referenced other types of frameworks used in their evaluation. This review summarizes how factors relevant to different aspects of implementation within the included papers could have been organized using the Consolidated Framework for Implementation Research. The findings from this review highlight an opportunity for standardization to better inform the evidence for and delivery of sexual and reproductive health interventions in humanitarian settings. Increased use of implementation research frameworks such as a modified Consolidated Framework for Implementation Research could work toward both expanding the evidence base and increasing standardization.

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Over the past few decades, the field of public health implementation research (IR) has grown as a means by which the real-world conditions affecting health improvement efforts can be better understood. Peters et al. put forward the following broad definition of IR for health: “IR is the scientific inquiry into questions concerning implementation – the act of carrying an intention into effect, which in health research can be policies, programmes, or individual practices (collectively called interventions)” [ 1 ].

As IR emphasizes real-world circumstances, the context within which a health intervention is delivered is a core consideration. However, much IR implemented to date has focused on higher-resource settings, with many proposed frameworks developed with particular utility for a higher-income setting [ 2 ]. In recognition of IR’s potential to increase evidence across a range of settings, there have been numerous reviews of the use of IR in lower-resource settings as well as calls for broader use [ 3 , 4 ]. There have also been more focused efforts to modify various approaches and frameworks to strengthen the relevance of IR to low- and middle-income country settings (LMICs), such as the work by Means et al. to adapt a specific IR framework for increased utility in LMICs [ 2 ].

Within LMIC settings, the centrality of context to a health intervention’s impact is of particular relevance in humanitarian settings, which present a set of distinct implementation challenges [ 5 ]. Humanitarian responses to crisis situations operate with limited resources, under potential security concerns, and often under pressure to relieve acute suffering and need [ 6 ]. Given these factors, successful implementation of a particular health intervention may require different qualities than those that optimize intervention impact under more stable circumstances [ 7 ]. Despite increasing recognition of the need for expanded evidence of health interventions in humanitarian settings, the evidence base remains limited [ 8 ]. Furthermore, despite its potential utility, there is not standardized guidance on IR in humanitarian settings, nor are there widely endorsed recommendations for the frameworks best suited to analyze implementation in these settings.

Sexual and reproductive health (SRH) is a core aspect of the health sector response in humanitarian settings [ 9 ]. Yet, progress in addressing SRH needs has lagged far behind other services because of challenges related to culture and ideology, financing constraints, lack of data and competing priorities [ 10 ]. The Minimum Initial Service Package (MISP) for SRH in Crisis Situations is the international standard for the minimum set of SRH services that should be implemented in all crisis situations [ 11 ]. However, as in other areas of health, there is need for expanded evidence for planning and implementation of SRH interventions in humanitarian settings. Recent systematic reviews of SRH in humanitarian settings have focused on the effectiveness of interventions and service delivery strategies, as well as factors affecting utilization, but have not detailed whether IR frameworks were used [ 12 , 13 , 14 , 15 ]. There have also been recent reviews examining IR frameworks used in various settings and research areas, but none have explicitly focused on humanitarian settings [ 2 , 16 ].

Given the need for an expanded evidence base for SRH interventions in humanitarian settings and the potential for IR to be used to expand the available evidence, a scoping review was undertaken. This scoping review sought to identify IR approaches that have been used in the last ten years to evaluate SRH interventions in humanitarian settings.

This review also sought to shed light on whether there is a need for a common framework to guide research design, analysis, and reporting for SRH interventions in humanitarian settings and if so, if there are any established frameworks already in use that would be fit-for-purpose or could be tailored to meet this need.

The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) extension for scoping reviews was utilized to guide the elements of this review [ 17 ]. The review protocol was retrospectively registered with the Open Science Framework ( https://osf.io/b5qtz ).

Search strategy

A two-fold search strategy was undertaken for this review, which covered the last 10 years (2013–2022). First, recent systematic reviews pertaining to research or evaluation of SRH interventions in humanitarian settings were identified through keyword searches on PubMed and Google Scholar. Four relevant systematic reviews were identified [ 12 , 13 , 14 , 15 ] Table 1 .

Second, a literature search mirroring these reviews was conducted to identify relevant papers published since the completion of searches for the most recent review (April 2017). Additional file 1 includes the search terms that were used in the literature search [see Additional file 1 ].

The literature search was conducted for papers published from April 2017 to December 2022 in the databases that were searched in one or more of the systematic reviews: PubMed, Embase, PsycInfo, CINAHL and Global Health. Searches were completed in January 2023 Table 2 .

Two reviewers screened each identified study for alignment with inclusion criteria. Studies in the four systematic reviews identified were considered potentially eligible if published during the last 10 years. These papers then underwent full-text review to confirm satisfaction of all inclusion criteria, as inclusion criteria were similar but not fully aligned across the four reviews.

Literature search results were exported into a citation manager (Covidence), duplicates were removed, and a step-wise screening process for inclusion was applied. First, all papers underwent title and abstract screening. The remaining papers after abstract screening then underwent full-text review to confirm satisfaction of all inclusion criteria. Title and abstract screening as well as full-text review was conducted independently by both authors; disagreements after full-text review were resolved by consensus.

Data extraction and synthesis

The following content areas were summarized in Microsoft Excel for each paper that met inclusion criteria: publication details including author, year, country, setting [rural, urban, camp, settlement], population [refugees, internally displaced persons, general crisis-affected], crisis type [armed conflict, natural disaster], crisis stage [acute, chronic], study design, research methods, SRH intervention, and intervention target population [specific beneficiaries of the intervention within the broader population]; the use of an IR framework; details regarding the IR framework, how it was used, and any rationale given for the framework used; factors cited as impacting SRH interventions, either positively or negatively; and other key findings deemed relevant to this review.

As the focus of this review was on the approach taken for SRH intervention research and evaluation, the quality of the studies themselves was not assessed.

Twenty papers underwent full-text review due to their inclusion in one or more of the four systematic reviews and meeting publication date inclusion criteria. The literature search identified 7,016 unique papers. After full-text screening, 69 met all inclusion criteria and were included in the review. Figure  1 illustrates the search strategy and screening process.

Flow chart of paper identification

Papers published in each of the 10 years of the review timeframe (2013–2022) were included. 29% of the papers originated from the first five years of the time frame considered for this review, with the remaining 71% papers coming from the second half. Characteristics of included publications, including geographic location, type of humanitarian crisis, and type of SRH intervention, are presented in Table  3 .

A wide range of study designs and methods were used across the papers, with both qualitative and quantitative studies well represented. Twenty-six papers were quantitative evaluations [ 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 ], 17 were qualitative [ 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 ], and 26 used mixed methods [ 61 , 62 , 63 , 64 , 65 , 66 , 67 , 68 , 69 , 70 , 71 , 72 , 73 , 74 , 75 , 76 , 77 , 78 , 79 , 80 , 81 , 82 , 83 , 84 , 85 , 86 ]. Within the quantitative evaluations, 15 were observational, while five were quasi-experimental, five were randomized controlled trials, and one was an economic evaluation. Study designs as classified by the authors of this review are summarized in Table  4 .

Six papers (9%) explicitly cited use of an IR framework. Three of these papers utilized the Consolidated Framework for Implementation Research (CFIR) [ 51 , 65 , 70 ]. The CFIR is a commonly used determinant framework that—in its originally proposed form in 2009—is comprised of five domains, each of which has constructs to further categorize factors that impact implementation. The CFIR domains were identified as core content areas influencing the effectiveness of implementation, and the constructs within each domain are intended to provide a range of options for researchers to select from to “guide diagnostic assessments of implementation context, evaluate implementation progress, and help explain findings.” [ 87 ] To allow for consistent terminology throughout this review, the original 2009 CFIR domains and constructs are used.

Guan et al. conducted a mixed methods study to assess the feasibility and effectiveness of a neonatal hepatitis B immunization program in a conflict-affected rural region of Myanmar. Guan et al. report mapping data onto the CFIR as a secondary analysis step. They describe that “CFIR was used as a comprehensive meta-theoretical framework to examine the implementation of the Hepatitis B Virus vaccination program,” and implementation themes from multiple study data sources (interviews, observations, examination of monitoring materials) were mapped onto CFIR constructs. They report their results in two phases – Pre-implementation training and community education, and Implementation – with both anchored in themes that they had mapped onto CFIR domains and constructs. All but six constructs were included in their analysis, with a majority summarized in a table and key themes explored further in the narrative text. They specify that most concerns were identified within the Outer Setting and Process domains, while elements identified within the Inner Setting domain provided strength to the intervention and helped mitigate against barriers [ 70 ].

Sarker et al. conducted a qualitative study to assess provision of maternal, newborn and child health services to Rohingya refugees residing in camps in Cox’s Bazar, Bangladesh. They cite using CFIR as a guide for thematic analysis, applying it after a process of inductive and deductive coding to index these codes into the CFIR domains. They utilized three of the five CFIR domains (Outer Setting, Inner Setting, and Process), stating that the remaining two domains (Intervention Characteristics and Characteristics of Individuals) were not relevant to their analysis. They then proposed two additional CFIR domains, Context and Security, for use in humanitarian contexts. In contrast to Guan et al., CFIR constructs are not used nor mentioned by Sarker et al., with content under each domain instead synthesized as challenges and potential solutions. Regarding the CFIR, Sarker et al. write, “The CFIR guided us for interpretative coding and creating the challenges and possible solutions into groups for further clarification of the issues related to program delivery in a humanitarian crisis setting.” [ 51 ]

Sami et al. conducted a mixed methods case study to assess the implementation of a package of neonatal interventions at health facilities within refugee and internally displaced persons camps in South Sudan. They reference use of the CFIR earlier in the study than Sarker et al., basing their guides for semi-structured focus group discussions on the CFIR framework. They similarly reference a general use of the CFIR framework as they conducted thematic analysis. Constructs are referenced once, but they do not specify whether their application of the CFIR framework included use of domains, constructs, or both. This may be in part because they then applied an additional framework, the World Health Organization (WHO) Health System Framework, to present their findings. They describe a nested approach to their use of these frameworks: “Exploring these [CFIR] constructs within the WHO Health Systems Framework can identify specific entry points to improve the implementation of newborn interventions at critical health system building blocks.” [ 65 ]

Three papers cite use of different IR frameworks. Bolan et al. utilized the Theoretical Domains Framework in their mixed methods feasibility study and pilot cluster randomized trial evaluating pilot use of the Safe Delivery App by maternal and newborn health workers providing basic emergency obstetric and newborn care in facilities in the conflict-affected Maniema province of the Democratic Republic of the Congo (DRC). They used the Theroetical Domains Framework in designing interview questions, and further used it as the coding framework for their analysis. Similar to the CFIR, the Theoretical Domains Framework is a determinant framework that consists of domains, each of which then includes constructs. Bolan et al. utilized the Theoretical Domains Framework at the construct level in interview question development and at the domain level in their analysis, mapping interview responses to eight of the 14 domains [ 83 ]. Berg et al. report using an “exploratory design guided by the principles of an evaluation framework” developed by the Medical Research Council to analyze the implementation process, mechanisms of impact, and outcomes of a three-pillar training intervention to improve maternal and neonatal healthcare in the conflict-affected South Kivu province of the DRC [ 67 , 88 ]. Select components of this evaluation framework were used to guide deductive analysis of focus group discussions and in-depth interviews [ 67 ]. In their study of health workers’ knowledge and attitudes toward newborn health interventions in South Sudan, before and after training and supply provision, Sami et al. report use of the Conceptual Framework of the Role of Attitudes in Evidence-Based Practice Implementation in their analysis process. The framework was used to group codes following initial inductive coding analysis of in-depth interviews [ 72 ].

Three other papers cite use of specific frameworks in their intervention evaluation [ 19 , 44 , 76 ]. As a characteristic of IR is the use of an explicit framework to guide the research, the use of the frameworks in these three papers meets the intention of IR and serves the purpose that an IR framework would have in strengthening the analytical rigor. Castle et al. cite use of their program’s theory of change as a framework for a mixed methods evaluation of the provision of family planning services and more specifically uptake of long-acting reversible contraception use in the DRC. They describe use of the theory of change to “enhance effectiveness of [long-acting reversible contraception] access and uptake.” [ 76 ] Thommesen et al. cite use of the AAAQ (Availability, Accessibility, Acceptability and Quality) framework in their qualitative study assessing midwifery services provided to pregnant women in Afghanistan. This framework is focused on the “underlying elements needed for attainment of optimum standard of health care,” but the authors used it in this paper to evaluate facilitators and barriers to women accessing midwifery services [ 44 ]. Jarrett et al. cite use of the Centers for Disease Control and Prevention’s (CDC) Guidelines for Evaluating Public Health Surveillance Systems to explore the characteristics of a population mobility, mortality and birth surveillance system in South Kivu, DRC. Use of these CDC guidelines is cited as one of four study objectives, and commentary is included in the Results section pertaining to each criteria within these guidelines, although more detail regarding use of these guidelines or the authors’ experience with their use in the study is not provided [ 19 ].

Overall, 22 of the 69 papers either explicitly or implicitly identified IR as relevant to their work. Nineteen papers include a focus on feasibility (seven of which did not otherwise identify the importance of exploring questions concerning implementation), touching on a common outcome of interest in implementation research [ 89 ].

While a majority of papers did not explicitly or implicitly use an IR framework to evaluate their SRH intervention of focus, most identified factors that facilitated implementation when they were present or served as a barrier when absent. Sixty cite factors that served as facilitators and 49 cite factors that served as barriers, with just three not citing either. Fifty-nine distinct factors were identified across the papers.

Three of the six studies that explicitly used an IR framework used the CFIR, and the CFIR is the only IR framework that was used by multiple studies. As previously mentioned, Means et al. put forth an adaptation of the CFIR to increase its relevance in LMIC settings, proposing a sixth domain (Characteristics of Systems) and 11 additional constructs [ 2 ]. Using the expanded domains and constructs as proposed by Means et al., the 59 factors cited by papers in this review were thematically grouped into the six domains: Characteristics of Systems, Outer Setting, Inner Setting, Characteristics of Individuals, Intervention Characteristics, and Process. Within each domain, alignment with CFIR constructs was assessed for, and alignment was found with 29 constructs: eight of Means et al.’s 11 constructs, and 21 of the 39 standard CFIR constructs. Three factors did not align with any construct (all fitting within the Outer Setting domain), and 14 aligned with a construct label but not the associated definition. Table 5 synthesizes the mapping of factors affecting SRH intervention implementation to CFIR domains and constructs, with the construct appearing in italics if it is considered to align with that factor by label but not by definition.

Table 6 lists the CFIR constructs that were not found to have alignment with any factor cited by the papers in this review.

This scoping review sought to assess how IR frameworks have been used to bolster the evidence base for SRH interventions in humanitarian settings, and it revealed that IR frameworks, or an explicit IR approach, are rarely used. All four of the systematic reviews identified with a focus on SRH in humanitarian settings articulate the need for more research examining the effectiveness of SRH interventions in humanitarian settings, with two specifically citing a need for implementation research/science [ 12 , 13 ]. The distribution of papers across the timeframe included in this review does suggest that more research on SRH interventions for crisis-affected populations is taking place, as a majority of relevant papers were published in the second half of the review period. The papers included a wide range of methodologies, which reflect the differing research questions and contexts being evaluated. However, it also invites the question of whether there should be more standardization of outcomes measured or frameworks used to guide analysis and to facilitate increased comparison, synthesis and application across settings.

Three of the six papers that used an IR framework utilized the CFIR. Guan et al. used the CFIR at both a domain and construct level, Sarker et al. used the CFIR at the domain level, and Sami et al. did not specify which CFIR elements were used in informing the focus group discussion guide [ 51 , 65 , 70 ]. It is challenging to draw strong conclusions about the applicability of CFIR in humanitarian settings based on the minimal use of CFIR and IR frameworks within the papers reviewed, although Guan et al. provides a helpful model for how analysis can be structured around CFIR domains and constructs. It is worth considering that the minimal use of IR frameworks, and more specifically CFIR constructs, could be in part because that level of prescriptive categorization does not allow for enough fluidity in humanitarian settings. It also raises questions about the appropriate degree of standardization to pursue for research done in these settings.

The mapping of factors affecting SRH intervention implementation provides an example of how a modified CFIR framework could be used for IR in humanitarian contexts. This mapping exercise found factors that mapped to all five of the original CFIR domains as well as the sixth domain proposed by Means et al. All factors fit well within the definition for the selected domain, indicating an appropriate degree of fit between these existing domains and the factors identified as impacting SRH interventions in humanitarian settings. On a construct level, however, the findings were more variable, with one-quarter of factors not fully aligning with any construct. Furthermore, over 40% of the CFIR constructs (including the additional constructs from Means et al.) were not found to align with any factors cited by the papers in this review, also demonstrating some disconnect between the parameters posed by the CFIR constructs and the factors cited as relevant in a humanitarian context.

It is worth noting that while the CFIR as proposed in 2009 was used in this assessment, as well as in the included papers which used the CFIR, an update was published in 2022. Following a review of CFIR use since its publication, the authors provide updates to construct names and definitions to “make the framework more applicable across a range of innovations and settings.” New constructs and subconstructs were also added, for a total of 48 constructs and 19 subconstructs across the five domains [ 90 ]. A CFIR Outcomes Addendum was also published in 2022, based on recommendations for the CFIR to add outcomes and intended to be used as a complement to the CFIR determinants framework [ 91 ]. These expansions to the CFIR framework may improve applicability of the CFIR in humanitarian settings. Several constructs added to the Outer Setting domain could be of particular utility – critical incidents, local attitudes, and local conditions, each of which could help account for unique challenges faced in contexts of crisis. Sub-constructs added within the Inner Setting domain that seek to clarify structural characteristics and available resources would also be of high utility based on mapping of the factors identified in this review to the original CFIR constructs. As outcomes were not formally included in the CFIR until the 2022 addendum, a separate assessment of implementation outcomes was not undertaken in this review. However, analysis of the factors cited by papers in this review as affecting implementation was derived from the full text of the papers and thus captures content relevant to implementation determinants that is contained within the outcomes.

Given the demonstrated need for additional flexibility within an IR framework for humanitarian contexts, while not a focus of this review, it is worth considering whether a different framework could provide a better fit than the CFIR. Other frameworks have differing points of emphasis that would create different opportunities for flexibility but that do not seem to resolve the challenges experienced in applying the CFIR to a humanitarian context. As one example, the EPIS (Exploration, Preparation, Implementation, Sustainment) Framework considers the impact of inner and outer context on each of four implementation phases; while the constructs within this framework are broader than the CFIR, an emphasis on the intervention characteristics is missing, a domain where stronger alignment within the CFIR is also needed [ 92 ]. Alternatively, the PRISM (Practical, Robust Implementation and Sustainability Model) framework is a determinant and evaluation framework that adds consideration of context factors to the RE-AIM (Reach, Effectiveness, Adoption, Implementation, Maintenance) outcomes framework. It has a stronger emphasis on intervention aspects, with sub-domains to account for both organization and patient perspectives within the intervention. While PRISM does include aspects of context, external environment considerations are less robust and intentionally less comprehensive in scope, which would not provide the degree of alignment possible between the Characteristics of Systems and Outer Setting CFIR domains for the considerations unique to humanitarian environments [ 93 ].

Reflecting on their experience with the CFIR, Sarker et al. indicate that it can be a “great asset” in both evaluating current work and developing future interventions. They also encourage future research of humanitarian health interventions to utilize the CFIR [ 51 ]. The other papers that used the CFIR do not specifically reflect on their experience utilizing it, referring more generally to having felt that it was a useful tool [ 65 , 70 ]. On their use of an evaluation framework, Berg et al. reflected that it lent useful structure and helped to identify aspects affecting implementation that otherwise would have gone un-noticed [ 67 ]. The remaining studies that utilized an IR framework did not specifically comment on their experience with its use [ 72 , 83 ]. While a formal IR framework was not engaged by other studies, a number cite a desire for IR to contribute further detail to their findings [ 21 , 37 ].

In their recommendations for strengthening the evidence base for humanitarian health interventions, Ager et al. speak to the need for “methodologic innovation” to develop methodologies with particular applicability in humanitarian settings [ 7 ]. As IR is not yet routinized for SRH interventions, this could be opportune timing for the use of a standardized IR framework to gauge its utility. Using an IR framework to assess factors influencing implementation of the MISP in initial stages of a humanitarian response, and interventions to support more comprehensive SRH service delivery in protracted crises, could lend further rigor and standardization to SRH evaluations, as well as inform strategies to improve MISP implementation over time. Based on categorizing factors identified by these papers as relevant for intervention evaluation, there does seem to be utility to a modified CFIR approach. Given the paucity of formal IR framework use within SRH literature, it would be worth conducting similar scoping exercises to assess for explicit use of IR frameworks within the evidence base for other health service delivery areas in humanitarian settings. In the interim, the recommended approach from this review for future IR on humanitarian health interventions would be a modified CFIR approach with domain-level standardization and flexibility for constructs that may standardize over time with more use. This would enable use of a common analytical framework and vocabulary at the domain level for stakeholders to describe interventions and the factors influencing the effectiveness of implementation, with constructs available to use and customize as most appropriate for specific contexts and interventions.

This review had a number of limitations. As this was a scoping review and a two-part search strategy was used, the papers summarized here may not be comprehensive of those written pertaining to SRH interventions over the past 10 years. Papers from 2013 to 2017 that would have met this scoping review’s inclusion criteria may have been omitted due to being excluded from the systematic reviews. The review was limited to papers available in English. Furthermore, this review did not assess the quality of the papers included or seek to assess the methodology used beyond examination of the use of an IR framework. It does, however, serve as a first step in assessing the extent to which calls for implementation research have been addressed, and identify entry points for strengthening the science and practice of SRH research in humanitarian settings.

With one in 23 people worldwide in need of humanitarian assistance, and financing required for response plans at an all-time high, the need for evidence to guide resource allocation and programming for SRH in humanitarian settings is as important as ever [ 94 ]. Recent research agenda setting initiatives and strategies to advance health in humanitarian settings call for increased investment in implementation research—with priorities ranging from research on effective strategies for expanding access to a full range of contraceptive options to integrating mental health and psychosocial support into SRH programming to capturing accurate and actionable data on maternal and perinatal mortality in a wide range of acute and protracted emergency contexts [ 95 , 96 ]. To truly advance guidance in these areas, implementation research will need to be conducted across diverse humanitarian settings, with clear and consistent documentation of both intervention characteristics and outcomes, as well as contextual and programmatic factors affecting implementation.

Conclusions

Implementation research has potential to increase impact of health interventions particularly in crisis-affected settings where flexibility, adaptability and context-responsive approaches are highlighted as cornerstones of effective programming. There remains significant opportunity for standardization of research in the humanitarian space, with one such opportunity occurring through increased utilization of IR frameworks such as a modified CFIR approach. Investing in more robust sexual and reproductive health research in humanitarian contexts can enrich insights available to guide programming and increase transferability of learning across settings.

Availability of data and materials

The datasets analyzed during the current study are available from the corresponding author on reasonable request.

Abbreviations

Availability, Accessibility, Acceptability and Quality

Centers for Disease Control and Prevention

Consolidated Framework for Implementation Research

Democratic Republic of the Congo

Exploration, Preparation, Implementation, Sustainment

• Implementation research

Low and middle income country

Minimum Initial Service Package

Practical, Robust Implementation and Sustainability Model

Preferred Reporting Items for Systematic Reviews and Meta-Analyses

Reach, Effectiveness, Adoption, Implementation, Maintenance

• Sexual and reproductive health

World Health Organization

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Financial hardship among patients suffering from neglected tropical diseases: A systematic review and meta-analysis of global literature

Roles Conceptualization, Data curation, Formal analysis, Funding acquisition, Investigation, Methodology, Project administration, Resources, Software, Visualization, Writing – original draft

Affiliations Department of Pharmacotherapy, College of Pharmacy, University of Utah, Salt Lake City, Utah, United States of America, Department of Social and Administrative Pharmacy, Faculty of Pharmaceutical Sciences, Chulalongkorn University, Bangkok, Thailand

Roles Conceptualization, Data curation, Formal analysis, Investigation, Validation, Writing – review & editing

Affiliations Department of Pharmacotherapy, College of Pharmacy, University of Utah, Salt Lake City, Utah, United States of America, School of Pharmacy, Sungkyunkwan University, Suwon, South Korea

Roles Investigation, Writing – review & editing

Affiliation Corvaxan Foundation, Villanova, Pennsylvania, United States of America

Roles Conceptualization, Writing – review & editing

Affiliation Department of Global Programme for Neglected Tropical Diseases, World Health Organization, Geneva, Switzerland

Roles Conceptualization, Funding acquisition, Project administration, Writing – review & editing

* E-mail: [email protected]

Affiliations Department of Pharmacotherapy, College of Pharmacy, University of Utah, Salt Lake City, Utah, United States of America, IDEAS Center, Veterans Affairs Salt Lake City Healthcare System, Salt Lake City, Utah, United States of America

• Chanthawat Patikorn, 
• Jeong-Yeon Cho, 
• Joshua Higashi, 
• Xiao Xian Huang, 
• Nathorn Chaiyakunapruk

• Published: May 13, 2024
• https://doi.org/10.1371/journal.pntd.0012086
• Peer Review
• Reader Comments

Introduction

Neglected tropical diseases (NTDs) mainly affect underprivileged populations, potentially resulting in catastrophic health spending (CHS) and impoverishment from out-of-pocket (OOP) costs. This systematic review aimed to summarize the financial hardship caused by NTDs.

We searched PubMed, EMBASE, EconLit, OpenGrey, and EBSCO Open Dissertations, for articles reporting financial hardship caused by NTDs from database inception to January 1, 2023. We summarized the study findings and methodological characteristics. Meta-analyses were performed to pool the prevalence of CHS. Heterogeneity was evaluated using the I 2 statistic.

Ten out of 1,768 studies were included, assessing CHS (n = 10) and impoverishment (n = 1) among 2,761 patients with six NTDs (Buruli ulcer, chikungunya, dengue, visceral leishmaniasis, leprosy, and lymphatic filariasis). CHS was defined differently across studies. Prevalence of CHS due to OOP costs was relatively low among patients with leprosy (0.0–11.0%), dengue (12.5%), and lymphatic filariasis (0.0–23.0%), and relatively high among patients with Buruli ulcers (45.6%). Prevalence of CHS varied widely among patients with chikungunya (11.9–99.3%) and visceral leishmaniasis (24.6–91.8%). Meta-analysis showed that the pooled prevalence of CHS due to OOP costs of visceral leishmaniasis was 73% (95% CI; 65–80%, n = 2, I 2 = 0.00%). Costs of visceral leishmaniasis impoverished 20–26% of the 61 households investigated, depending on the costs captured. The reported costs did not capture the financial burden hidden by the abandonment of seeking healthcare.

NTDs lead to a substantial number of households facing financial hardship. However, financial hardship caused by NTDs was not comprehensively evaluated in the literature. To develop evidence-informed strategies to minimize the financial hardship caused by NTDs, studies should evaluate the factors contributing to financial hardship across household characteristics, disease stages, and treatment-seeking behaviors.

Author summary

Neglected tropical diseases (NTDs) mainly affect underprivileged populations, potentially resulting in catastrophic health spending (CHS) and impoverishment from out-of-pocket (OOP) costs. This systematic review aimed to summarize the financial hardship caused by NTDs. We found that NTDs lead to a substantial number of households facing financial hardship. CHS risk due to direct OOP costs was relatively low among patients with leprosy (0.0–11.0%), dengue (12.5%), and lymphatic filariasis (0.0–23.0%), and relatively high among patients with Buruli ulcers (45.6%). CHS risk varied widely among patients with chikungunya (11.9–99.3%) and visceral leishmaniasis (24.6–91.8%). Costs of visceral leishmaniasis impoverished 20–26% of 61 households, depending on the costs captured. Nevertheless, financial hardship caused by NTDs was not comprehensively evaluated in the literature. Therefore, to develop evidence-informed strategies to minimize the financial hardship caused by NTDs, studies should evaluate the factors contributing to financial hardship across household characteristics, disease stages, and treatment-seeking behaviors.

Citation: Patikorn C, Cho J-Y, Higashi J, Huang XX, Chaiyakunapruk N (2024) Financial hardship among patients suffering from neglected tropical diseases: A systematic review and meta-analysis of global literature. PLoS Negl Trop Dis 18(5): e0012086. https://doi.org/10.1371/journal.pntd.0012086

Editor: Yoel Lubell, Mahidol-Oxford Tropical Medicine Research Unit, THAILAND

Received: November 7, 2023; Accepted: March 20, 2024; Published: May 13, 2024

Copyright: © 2024 Patikorn et al. This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Data Availability: The authors confirm that all data underlying the findings are fully available without restriction. All relevant data are within the paper and its Supporting Information files.

Funding: This study is funded by the Department of Control of Neglected Tropical Diseases, World Health Organization, Geneva, Switzerland. XXH, as an employee of the World Health Organization, contributed to this study in terms of study design, data interpretation, and report writing.

Competing interests: I have read the journal’s policy and the authors of this manuscript have the following competing interests:XXH works for the World Health Organization. The author alone is responsible for the views expressed in this publication and does not necessarily represent the decisions, policies, or views of the World Health Organization.

In 2021, the World Health Organization (WHO) reported that 1.65 billion people required treatment and care for neglected tropical diseases (NTDs) as they faced humanistic, social, and economic burdens incurred by the diseases. NTDs are a diverse group of diseases that mainly affect underprivileged communities in tropical and subtropical areas [ 1 ]. NTDs predominantly affect disadvantaged populations in low- and middle-income countries (LMICs) due to the lack of timely access to affordable care. It has been reported that every low-income country is affected by at least five NTDs [ 2 ]. Even worse, impoverishment serves as a structural determinant. At the same time, it is a consequence of NTDs due to the direct and indirect costs incurred [ 3 ]. Therefore, the WHO has advocated in their recent NTDs 2021–2023 roadmap that NTDs must be overcome to attain Sustainable Development Goals (SDGs) and ensure Universal Health Coverage (UHC). The NTDs 2021–2030 roadmap targets that 90% of the population at risk are protected against catastrophic out-of-pocket (OOP) health spending caused by NTDs [ 1 ].

Financial hardship is usually quantified as catastrophic health spending (CHS) (as known as catastrophic health expenditure) and impoverishment. CHS is the proportion of households with OOP costs incurred by a specific disease that exceed a specific threshold of the total household income or expenditure (budget share approach) or non-subsistent household expenditure (capacity-to-pay approach). Impoverishment is when the OOP costs push households below the poverty line [ 4 – 6 ]. CHS and impoverishment are well-established indicators for the financial risk protection of the healthcare system, which was an essential dimension of the UHC as indicated under the SDG 3.8.2 indicators [ 1 , 7 ].

Financial hardship poses a greater challenge for individuals affected by NTDs, as they frequently reside in poverty before the onset of the disease. To evaluate the long-term economic risk imposed by health spending on NTDs, it is important to understand the coping strategies of this population. Literature has shown that coping strategies, such as seeking financial assistance through loans or selling their assets, could push households into or further into poverty if it impacts their productivity [ 8 ]. Thus, providing coverage to these groups effectively strengthens the financial risk protection of the health system [ 7 ]. Since some types of NTD are closely related to financial hardship, improving their financial protection may help attain UHC, especially for LMICs [ 9 ].

Financial protection is an essential indicator for NTDs and UHC; however, there was limited research on the financial hardship of NTDs. Although many studies addressed the question of the economic burden of NTDs, there is no systematic review and meta-analysis summarizing the financial hardship faced by the population affected by NTDs. Therefore, to fill this knowledge gap and build a baseline for the NTDs roadmap’s financial risk protection indicator, this study aimed to summarize the prevalence and magnitude of financial hardship among patients suffering from NTDs. Additionally, we assessed the methodologies of quantifying CHS and impoverishment incurred by NTDs.

Scope of the review

The protocol of this systematic review was registered with PROSPERO (CRD42023385627) [ 10 ]. This study was reported following the 2020 Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) reporting guideline ( S1 PRISMA Checklist) [ 11 ]. Differences from the original review protocol are described with rationale ( S1 Table ).

This systematic literature review focused on 20 diseases selected as NTDs by WHO: Buruli ulcer, Chagas disease, dengue and chikungunya, dracunculiasis (Guinea-worm disease), echinococcosis, foodborne trematodiases, human African trypanosomiasis (sleeping sickness), leishmaniasis, leprosy (Hansen’s disease), lymphatic filariasis, mycetoma, chromoblastomycosis and other deep mycoses, onchocerciasis (river blindness), rabies, scabies and other ectoparasitoses, schistosomiasis, soil-transmitted helminthiases, snakebite envenoming, taeniasis/cysticercosis, trachoma, and yaws and other endemic treponematoses [ 12 ].

Outcomes of interest of this systematic review were the prevalence and magnitude of victims who faced financial hardship caused by NTDs, including CHS, impoverishment, and coping strategies.

Search strategy and selection process

We searched three bibliographic databases, PubMed, EMBASE, and EconLit, to identify articles reporting financial hardship among patients suffering from NTDs from any country indexed from database inception to January 1, 2023. We also searched for grey literature in two databases, OpenGrey and EBSCO Open Dissertations. The search terms used were ( Disease name and its synonyms ) AND (catastroph* OR impoverish* OR coping OR economic consequence* OR out-of-pocket OR "out of pocket" OR ((household OR family OR patient AND (cost* OR spending OR expen*))), that was adapted to match the search techniques of each database. A full search strategy is shown in S2 Table . There was no language restriction applied in this systematic review. A supplemental search was performed by tracking citation and snowballing the eligible articles’ reference list.

Two reviewers (CP and JYC) independently performed the study selection. They screened the titles and abstracts of identified articles from database searches for relevance. Potentially relevant articles were sought for full-text articles. We requested the authors for full-text articles or reports of highly relevant articles without full-text articles, such as conference abstracts. The retrieved full-text articles were selected based on the eligibility criteria. Discrepancies arising during study selection were resolved by discussion with the third reviewer (NC).

Eligibility criteria

We included empirical studies reporting CHS, impoverishment, or coping strategies incurred by NTDs using primary data collection.

Data extraction

We developed a data extraction sheet by performing a pilot test of extracting five randomly selected articles and refining it until finalization. Two reviewers (CP and JYC) independently performed data extraction. Another reviewer (JH) checked the extracted data for correctness. Any discrepancies were resolved by discussion among reviewers.

Study findings and methodological characteristics extracted from the eligible articles are as follows: first author, publication year, NTDs, study setting, study design, sample characteristics, sample size, data collection period, data collection methods, time horizon, a perspective of the analysis, discount rate, costing year, reported currency, cost units, the definition of CHS and impoverishment, prevalence and magnitude of CHS and impoverishment incurred, economic consequences and coping strategies of financial hardship. Corresponding authors of the eligible articles were contacted to request individual patient-level data. However, we received no response.

The financial risk protection metric is intended to capture only the OOP costs for medical services (e.g., treatment and diagnosis costs). However, some studies considered certain types of direct non-medical costs (e.g., transportation, food, and accommodation costs) and indirect costs (e.g., productivity and income losses) when quantifying financial hardship. Some studies also included informal care costs, such as traditional medicine, as OOP costs [ 6 ]. Thus, our systematic review categorized costs extracted from the eligible studies as direct costs (OOP costs) and indirect costs. Direct costs were further categorized as direct medical costs and direct non-medical costs. The combination of direct costs and indirect costs was categorized as total costs.

Quality assessment

Two reviewers independently assessed the eligible articles’ quality (CP and JYC). Any discrepancies were resolved by consensus among the reviewers. To the best of our knowledge, there is no risk-of-bias assessment tool for economic burden studies. Hence, we assessed the quality of the eligible articles using the cost-of-illness evaluation checklist by Larg and Moss [ 13 ].

Data synthesis

A narrative synthesis was performed to summarize study findings, methodological characteristics, and the quality of the eligible studies. The identified countries were categorized based on the World Bank’s income levels and regions [ 14 ].

Statistical analysis

We performed meta-analyses to calculate the pooled prevalence of households experiencing financial hardship. However, this was possible only for studies that quantified financial hardship using the same measurement definition for a particular NTD. For example, we performed a meta-analysis to calculate the pooled prevalence of households experiencing CHS due to visceral leishmaniasis based on two studies that defined CHS as direct costs exceeding 10% of annual household income [ 8 , 15 ]. The remaining studies were not meta-analyzed due to the differences in the definitions of CHS. We estimated the pooled prevalence of CHS and 95% confidence intervals (CI) using a random-effects model under the DerSimonian and Laird approach [ 16 ]. Effect sizes were computed using each study’s Freeman–Tukey double-arcsine-transformed proportion. This variance-stabilizing transformation is particularly preferable when the proportions are close to 0 or 1 [ 17 ]. p < .05 was considered statistically significant in 2-sided tests.

Heterogeneity was evaluated by observing the forest plots and using the I 2 statistic that estimated the proportion of variability in a meta-analysis that is explained by differences between the included trials rather than by sampling error. Subgroup analyses were performed to explore possible causes of heterogeneity among study results. Publication bias was assessed using the funnel plot asymmetry test and the Egger regression asymmetry test [ 18 ]. Statistical analyses were conducted using Stata version 18.0 (Stata Corporation).

Patient and public involvement

Patients or the public were not involved in the design, or conduct, or reporting, or dissemination plans of our research.

Overall characteristics of the included studies

A total of 1,768 articles were identified from the search, of which 10 studies were included ( Fig 1 ) [ 8 , 15 , 19 – 26 ]. A list of excluded studies with reasons is presented in S3 Table . These studies quantified financial hardship among 2,761 patients in five LMICs (India, Nepal, Nigeria, Sudan, and Vietnam) who had been diagnosed with six out of the WHO’s 20 NTDs, including Buruli ulcer [ 20 ], chikungunya [ 21 , 26 ], dengue [ 22 ], visceral leishmaniasis [ 8 , 15 , 25 ], leprosy [ 19 , 23 ], and lymphatic filariasis [ 24 ]. Table 1 provides a summary of the study characteristics. We found no major concern in the quality of the included studies ( S4 Table )

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Financial hardship caused by NTDs was quantified as CHS (10 studies) [ 8 , 15 , 19 – 26 ], and impoverishment (1 study) [ 8 ]. All studies were conducted in LMICs with a focus on South Asia (7 studies) [ 8 , 19 , 21 , 23 – 26 ], Sub-Saharan Africa (2 studies) [ 15 , 20 ], East Asia & Pacific (1 study) [ 22 ]. Patients were mostly identified using a hospital-based approach (7 studies) [ 8 , 15 , 19 , 20 , 22 , 23 , 25 ], with active case-finding intervention implemented in two of those studies [ 20 , 23 ]. Five studies reported that patients sought informal healthcare, such as traditional healers, ayurveda, and homeopathy [ 19 – 21 , 25 , 26 ].

Costs captured in the financial hardship were direct medical costs (10 studies, 100%) [ 8 , 15 , 19 – 26 ], direct non-medical costs (9 studies, 90%) [ 8 , 15 , 19 – 21 , 23 – 26 ], and indirect costs (7 studies, 70%) [ 8 , 15 , 19 , 21 , 23 , 25 , 26 ], as summarized in Table 2 . These costs were captured with a different timeframe, including during a disease episode [ 8 , 15 , 20 , 21 , 25 , 26 ], during hospitalization in an intensive care unit [ 22 ], monthly costs with a maximum recall period of 3 years [ 19 ], per one outpatient visit in the last 6 months [ 23 ], and per one hospitalization episode in the last year and per one outpatient visit in the last 15 days [ 24 ]. Abandonment of healthcare seeking due to financial burden was not reflected in the reported costs as the included studies captured only patients who sought healthcare.

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The health insurance systems or special programs covered some of the costs. The costs for diagnosis and treatment of visceral leishmaniasis were provided free of charge to patients under the publicly financed health insurance system in Nepal [ 8 , 25 ] and Sudan [ 15 ]. In Nigeria, international development partners funded a special program that provided free diagnosis and treatment of Buruli ulcers, as well as accommodation, school funding, and basic allowance [ 20 ]. Additionally, the Indian government had a special program that provides financial assistance to families of patients affected by leprosy [ 19 ]. However, patients in India had to pay high OOP costs for medical services for leprosy [ 19 , 23 ], chikungunya [ 21 , 26 ], and lymphatic filariasis [ 24 ]. Similarly, patients in Vietnam also paid high OOP costs for the medical treatment of dengue [ 22 ]. For more details, refer to Table 3 .

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Financial hardship among patients suffering from NTDs

Catastrophic health spending..

CHS was variedly defined across studies in terms of types of costs (medical costs, medical and transportation costs, direct costs, indirect costs, or total costs), thresholds (5%, 10%, 15%, 25%, 30%, 40%, or 100%), timeframe (monthly, quarterly, or annual), household resources (income, consumption expenditure, national average annual household expenditure, or international poverty line) and perspective (household or individual). All studies used the budget share approach to quantify CHS. The most commonly used definitions of CHS caused by NTDs were direct costs of a disease episode exceeding 10% of annual household income (3 studies) [ 8 , 15 , 20 ] and total costs of a disease episode exceeding 10% of annual household income (3 studies) [ 8 , 15 , 25 ]. CHS that included only the direct medical costs was reported in two studies [ 8 , 22 ].

We summarized the prevalence of households experiencing CHS and the magnitude of CHS, determined as the percentage of the costs of NTDs as a share of income, in Table 4 . The prevalence and magnitude of CHS varied depending on the definitions of CHS, disease duration (episodic or chronic), and thresholds used (≤10% or >10%). Overall, the direct costs of NTDs resulted in a wide range of households experiencing CHS. CHS was generally low among patients with leprosy (0.0–11.0%) [ 19 , 23 ], dengue (12.5%) [ 22 ], and lymphatic filariasis (0.0–23.0%) [ 24 ], and relatively high among patients with Buruli ulcers (45.6%) [ 20 ]. CHS varied widely among patients with chikungunya (11.9–99.3%) [ 21 , 26 ] and visceral leishmaniasis (24.6–91.8%) [ 8 , 15 , 25 ].

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Meta-analyses were performed to pool the prevalence of CHS in studies reporting CHS using the same measurement definition in a particular CHS. This was only possible for visceral leishmaniasis, in which CHS was quantified as direct costs of a disease episode exceeding 10% of annual household income in two studies [ 8 , 15 ], and total costs exceeding 10% of annual household income in three studies [ 8 , 15 , 25 ].

The pooled prevalence of CHS, defined as direct costs exceeding 10% of annual household income, was 73% (95% CI; 65–80%, n = 2, I 2 = 0.00%), as shown in Fig 2A . Egger’s test (P = 0.80) indicated no evidence of small-study effects. Visual inspection of the funnel plot indicated no evidence of publication bias ( S1A Fig ).

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The pooled prevalence of CHS, defined as total costs exceeding 10% of annual household income, was 74% (95% CI; 49–93%, n = 3, I 2 = 94.72%), as shown in S2 Fig . We explored the source of heterogeneity by visual inspection of the forest plot. We found that the source of heterogeneity was the differences in the treatment of visceral leishmaniasis, where sodium stibogluconate was used in two studies [ 8 , 15 ], and miltefosine in one study [ 25 ]. Therefore, we performed a subgroup meta-analysis based on different treatments, as shown in Fig 2B . We removed one study [ 25 ] from the meta-analysis to investigate the publication bias without the presence of heterogeneity. Egger’s test (P = 0.81) indicated no evidence of small-study effects. Visual inspection of the funnel plot indicated no evidence of publication bias ( S1B Fig ).

Impoverishment.

Impoverishment was investigated in one study in patients with visceral leishmaniasis, which defined impoverishment as annual household income falling below the poverty line after paying for treatment [ 8 ]. Costs of visceral leishmaniasis impoverished 20–26% of the 61 households investigated, depending on the costs captured (20% medical costs, 21% medical and transportation costs, 26% direct costs), as shown in Table 2 .

Coping strategies

Four studies reported coping strategies used by patients to pay the costs of NTDs. These strategies included using savings (71–100% of patients), taking out loans (32–80%), selling livestock or other assets (17–32%), or borrowing money (0–23%), as shown in Table 2 . However, these studies did not distinguish between coping strategies used by patients who experienced CHS and those who did not [ 8 , 19 , 24 , 25 ].

Cost drivers and determinants of financial hardship

To understand the cost drivers of financial hardship caused by NTDs, we analyzed the percentage share of types of costs captured in the direct costs. The findings are presented in Fig 3 . Direct medical costs were the primary cost driver in nine studies [ 8 , 19 – 21 , 23 – 26 ]. However, one study identified food and transportation costs as the main cost drivers [ 15 ].

Abbreviation: ENL–erythema nodosum leprosum. Tripathy et al, 2020 [ 24 ]; Tiwari et al, 2018 [ 23 ]; Chandler et al, 2015 [ 19 ]; Uranw et al, 2013 [ 25 ], Meheus et al, 2013 [ 15 ], Adhikari et al, 2009 [ 8 ], McBride et al, 2019[ 22 ], Vijayakumar et al, 2013 [ 26 ], Gopalan et al, 2009 [ 21 ], Chukwu et al, 2017 [ 20 ] .

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Determinants of CHS were assessed in one study among patients with Buruli ulcers. The study concluded that neither age, gender, rural/urban location, education, occupation, religion, nor patient income group was a determinant of CHS [ 20 ]. There was no study investigating determinants of impoverishment.

NTDs primarily impact populations with limited financial means, yet the literature addressing the financial hardship caused by NTDs is relatively scarce. Our systematic review revealed that there were only ten studies covering six NTDs. We discovered that many households are facing financial hardship as a result of NTDs, despite having access to publicly funded healthcare systems or special NTD programs. The costs related to NTDs resulted in significant financial hardship for these households, mainly due to the high OOP costs associated with medical treatment. Even in situations where drugs used to treat NTDs were provided free of charge, the costs for supportive care, medical procedures, transportation, and food were still high and could have a devastating financial impact on these households. Moreover, these financial hardship indicators might not fully reflect the financial risk of the population affected by NTDs because many live in poverty or even extreme poverty. Victims of NTDs are usually those who are socially disadvantaged. They need to make trade-offs between suffering from the disease and seeking healthcare because not all victims can afford the costs of NTDs, especially OOP costs for medical treatment and transportation, which could lead to the abandonment of healthcare [ 1 – 3 ].

The research findings have shown that merely providing funding for treatments of NTDs is insufficient for protecting those affected by NTDs from financial hardship. Therefore, it is crucial to strengthen the entire healthcare system to effectively address the challenges of NTDs and provide financial protection to the victims. Additionally, it is important to encourage and engage communities to change the behavior of those affected by NTDs so that they seek medical assistance at appropriate healthcare facilities instead of relying on traditional healers or not seeking care at all. Our research also supports the need for an economic framework to guide NTD investments [ 27 ]. The ability to prioritize investments, informed partially by economic parameters, may appeal to a broad set of stakeholders and help facilitate the process of building coalitions to achieve the WHO’s goal that 90% of the at-risk population is protected against financial hardship caused by NTDs [ 1 ].

Although there is no consensus regarding the estimation approach and thresholds in quantifying CHS, it is important to note that these differences can significantly impact the findings and consequently impact the applications and implications of the findings [ 6 , 28 ]. We found that CHS was variedly defined across studies in terms of estimation approach, types of costs, thresholds, timeframe, household resources, and perspective. Our review revealed that 90% of the included studies captured direct non-medical costs as part of the OOP costs [ 8 , 15 , 19 – 21 , 23 – 26 ]. Furthermore, Seventy percent of the included studies considered indirect costs in quantifying financial hardship [ 8 , 15 , 19 , 21 , 23 , 25 , 26 ]. This approach aligned with an indicator called “catastrophic costs” that has emerged in tuberculosis studies. Catastrophic costs occur when the total healthcare costs, including direct and indirect costs, exceed 20% of the annual household income [ 28 ]. This indicator could be a more comprehensive measure of the overall financial burden of NTDs on the household beyond just the OOP costs which will be useful when evaluating and monitoring different healthcare policies and interventions to mitigate financial hardship caused by NTDs.

The findings of this systematic review and meta-analysis should be interpreted under the following limitations. The included studies in our review only focused on patients who sought healthcare, so the financial burden of those who did not seek healthcare was not captured in the reported OOP costs. This means that people who could not afford healthcare may have been excluded from these studies. Moreover, we could not perform meta-analyses of the prevalence of CHS on all identified NTDs due to differences in how CHS was quantified across studies and lack of access to individual patient-level data.

Hence, we highlighted some methodological considerations to guide future studies on financial hardship among households suffering from NTDs to gain a better understanding of the neglected public health issues and to inform the development of strategies of what to address to tackle the financial burden of NTDs. Firstly, methods to quantify financial hardship should be coherent to allow comparability across studies. For instance, CHS and impoverishment should be defined and measured in a relevant manner to the nature of the NTD, including estimation approach, thresholds, types of costs, timeframe, household resources, and perspective. Secondly, subgroup analyses should be conducted to evaluate the determinants of financial hardship across household characteristics (e.g., income, socioeconomic status) or phases of disease (e.g., disease onset, treatment seeking, diagnosis, treatment, post-treatment). Lastly, coping strategies should be assessed among those who did and did not experience financial hardship to understand the economic consequences of financial hardship across subgroups.

NTDs can be a devastating burden on households, not only in terms of physical and mental health but also financially. NTDs lead to a substantial number of households facing financial hardship. However, financial hardship caused by NTDs was not comprehensively evaluated in the literature. Furthermore, OOP costs represented only a partial picture of the financial hardship the population affected by NTDs faces. To mitigate this financial hardship, it is imperative to conduct thorough research to identify the factors contributing to it. Future research should consider various household characteristics, such as income, education level, and geographic location, as well as the different disease stages, from onset to treatment completion. Future studies should also investigate the hidden financial burden due to the abandonment of healthcare-seeking to capture the economic burden and opportunity costs of those who did not seek healthcare. By carefully examining these factors, researchers and decision-makers can gain insight into the specific challenges faced by households affected by NTDs and develop targeted interventions to alleviate financial hardships. Ultimately, these studies can help inform the development of strategies to reduce the burden of NTDs on households and improve overall health outcomes.

Supporting information

S1 prisma checklist. prisma checklist..

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S1 Table. Differences from original review protocol.

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S2 Table. Full search strategy.

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S3 Table. Excluded studies with reasons.

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S4 Table. Quality assessment using Larg, A., and Moss, J. R. (2011) Cost-of-illness studies: a guide to critical evaluation.

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S1 Fig. Assessment of publication bias.

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S2 Fig. Forest plot of pooled proportion of catastrophic health spending defined as total costs exceeding 10% of annual household income.

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Acknowledgments

The authors alone are responsible for the views expressed in this article and they do not necessarily represent the views, decisions or policies of the institutions with which they are affiliated.

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