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Research Guides

Multiple Case Studies

Nadia Alqahtani and Pengtong Qu

Description

The case study approach is popular across disciplines in education, anthropology, sociology, psychology, medicine, law, and political science (Creswell, 2013). It is both a research method and a strategy (Creswell, 2013; Yin, 2017). In this type of research design, a case can be an individual, an event, or an entity, as determined by the research questions. There are two variants of the case study: the single-case study and the multiple-case study. The former design can be used to study and understand an unusual case, a critical case, a longitudinal case, or a revelatory case. On the other hand, a multiple-case study includes two or more cases or replications across the cases to investigate the same phenomena (Lewis-Beck, Bryman & Liao, 2003; Yin, 2017). …a multiple-case study includes two or more cases or replications across the cases to investigate the same phenomena

The difference between the single- and multiple-case study is the research design; however, they are within the same methodological framework (Yin, 2017). Multiple cases are selected so that “individual case studies either (a) predict similar results (a literal replication) or (b) predict contrasting results but for anticipatable reasons (a theoretical replication)” (p. 55). When the purpose of the study is to compare and replicate the findings, the multiple-case study produces more compelling evidence so that the study is considered more robust than the single-case study (Yin, 2017).

To write a multiple-case study, a summary of individual cases should be reported, and researchers need to draw cross-case conclusions and form a cross-case report (Yin, 2017). With evidence from multiple cases, researchers may have generalizable findings and develop theories (Lewis-Beck, Bryman & Liao, 2003).

Creswell, J. W. (2013). Qualitative inquiry and research design: Choosing among five approaches (3rd ed.). Los Angeles, CA: Sage.

Lewis-Beck, M., Bryman, A. E., & Liao, T. F. (2003). The Sage encyclopedia of social science research methods . Los Angeles, CA: Sage.

Yin, R. K. (2017). Case study research and applications: Design and methods . Los Angeles, CA: Sage.

Key Research Books and Articles on Multiple Case Study Methodology

Yin discusses how to decide if a case study should be used in research. Novice researchers can learn about research design, data collection, and data analysis of different types of case studies, as well as writing a case study report.

Chapter 2 introduces four major types of research design in case studies: holistic single-case design, embedded single-case design, holistic multiple-case design, and embedded multiple-case design. Novice researchers will learn about the definitions and characteristics of different designs. This chapter also teaches researchers how to examine and discuss the reliability and validity of the designs.

Creswell, J. W., & Poth, C. N. (2017). Qualitative inquiry and research design: Choosing among five approaches . Los Angeles, CA: Sage.

This book compares five different qualitative research designs: narrative research, phenomenology, grounded theory, ethnography, and case study. It compares the characteristics, data collection, data analysis and representation, validity, and writing-up procedures among five inquiry approaches using texts with tables. For each approach, the author introduced the definition, features, types, and procedures and contextualized these components in a study, which was conducted through the same method. Each chapter ends with a list of relevant readings of each inquiry approach.

This book invites readers to compare these five qualitative methods and see the value of each approach. Readers can consider which approach would serve for their research contexts and questions, as well as how to design their research and conduct the data analysis based on their choice of research method.

Günes, E., & Bahçivan, E. (2016). A multiple case study of preservice science teachers’ TPACK: Embedded in a comprehensive belief system. International Journal of Environmental and Science Education, 11 (15), 8040-8054.

In this article, the researchers showed the importance of using technological opportunities in improving the education process and how they enhanced the students’ learning in science education. The study examined the connection between “Technological Pedagogical Content Knowledge” (TPACK) and belief system in a science teaching context. The researchers used the multiple-case study to explore the effect of TPACK on the preservice science teachers’ (PST) beliefs on their TPACK level. The participants were three teachers with the low, medium, and high level of TPACK confidence. Content analysis was utilized to analyze the data, which were collected by individual semi-structured interviews with the participants about their lesson plans. The study first discussed each case, then compared features and relations across cases. The researchers found that there was a positive relationship between PST’s TPACK confidence and TPACK level; when PST had higher TPACK confidence, the participant had a higher competent TPACK level and vice versa.

Recent Dissertations Using Multiple Case Study Methodology

Milholland, E. S. (2015). A multiple case study of instructors utilizing Classroom Response Systems (CRS) to achieve pedagogical goals . Retrieved from ProQuest Dissertations & Theses Global. (Order Number 3706380)

The researcher of this study critiques the use of Classroom Responses Systems by five instructors who employed this program five years ago in their classrooms. The researcher conducted the multiple-case study methodology and categorized themes. He interviewed each instructor with questions about their initial pedagogical goals, the changes in pedagogy during teaching, and the teaching techniques individuals used while practicing the CRS. The researcher used the multiple-case study with five instructors. He found that all instructors changed their goals during employing CRS; they decided to reduce the time of lecturing and to spend more time engaging students in interactive activities. This study also demonstrated that CRS was useful for the instructors to achieve multiple learning goals; all the instructors provided examples of the positive aspect of implementing CRS in their classrooms.

Li, C. L. (2010). The emergence of fairy tale literacy: A multiple case study on promoting critical literacy of children through a juxtaposed reading of classic fairy tales and their contemporary disruptive variants . Retrieved from ProQuest Dissertations & Theses Global. (Order Number 3572104)

To explore how children’s development of critical literacy can be impacted by their reactions to fairy tales, the author conducted a multiple-case study with 4 cases, in which each child was a unit of analysis. Two Chinese immigrant children (a boy and a girl) and two American children (a boy and a girl) at the second or third grade were recruited in the study. The data were collected through interviews, discussions on fairy tales, and drawing pictures. The analysis was conducted within both individual cases and cross cases. Across four cases, the researcher found that the young children’s’ knowledge of traditional fairy tales was built upon mass-media based adaptations. The children believed that the representations on mass-media were the original stories, even though fairy tales are included in the elementary school curriculum. The author also found that introducing classic versions of fairy tales increased children’s knowledge in the genre’s origin, which would benefit their understanding of the genre. She argued that introducing fairy tales can be the first step to promote children’s development of critical literacy.

Asher, K. C. (2014). Mediating occupational socialization and occupational individuation in teacher education: A multiple case study of five elementary pre-service student teachers . Retrieved from ProQuest Dissertations & Theses Global. (Order Number 3671989)

This study portrayed five pre-service teachers’ teaching experience in their student teaching phase and explored how pre-service teachers mediate their occupational socialization with occupational individuation. The study used the multiple-case study design and recruited five pre-service teachers from a Midwestern university as five cases. Qualitative data were collected through interviews, classroom observations, and field notes. The author implemented the case study analysis and found five strategies that the participants used to mediate occupational socialization with occupational individuation. These strategies were: 1) hindering from practicing their beliefs, 2) mimicking the styles of supervising teachers, 3) teaching in the ways in alignment with school’s existing practice, 4) enacting their own ideas, and 5) integrating and balancing occupational socialization and occupational individuation. The study also provided recommendations and implications to policymakers and educators in teacher education so that pre-service teachers can be better supported.

Multiple Case Studies Copyright © 2019 by Nadia Alqahtani and Pengtong Qu is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License , except where otherwise noted.

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Multiple Case Research Design

• First Online: 04 January 2024

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• Michael Blankenagel 3  

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This chapter addresses multiple case research designs’ peculiarities, characteristics, and major fallacies. The significant advantage of multiple-case research lies in cross-case analysis. A multiple-case research design shifts the focus from understanding a single case to the differences and similarities between cases. Thus, it is more than conducting more case studies (second, third, etc.). Instead, it is the next step in developing a theory about factors driving differences and similarities. Also, researchers find relevant information on how to write a multiple case research design paper and learn about typical methodologies used for this research design. The chapter closes by referring to overlapping and adjacent research designs.

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Hunziker, S., Blankenagel, M. (2024). Multiple Case Research Design. In: Research Design in Business and Management. Springer Gabler, Wiesbaden. https://doi.org/10.1007/978-3-658-42739-9_9

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Methodology

• What Is a Case Study? | Definition, Examples & Methods

What Is a Case Study? | Definition, Examples & Methods

Published on May 8, 2019 by Shona McCombes . Revised on November 20, 2023.

A case study is a detailed study of a specific subject, such as a person, group, place, event, organization, or phenomenon. Case studies are commonly used in social, educational, clinical, and business research.

A case study research design usually involves qualitative methods , but quantitative methods are sometimes also used. Case studies are good for describing , comparing, evaluating and understanding different aspects of a research problem .

Table of contents

When to do a case study, step 1: select a case, step 2: build a theoretical framework, step 3: collect your data, step 4: describe and analyze the case, other interesting articles.

A case study is an appropriate research design when you want to gain concrete, contextual, in-depth knowledge about a specific real-world subject. It allows you to explore the key characteristics, meanings, and implications of the case.

Case studies are often a good choice in a thesis or dissertation . They keep your project focused and manageable when you don’t have the time or resources to do large-scale research.

You might use just one complex case study where you explore a single subject in depth, or conduct multiple case studies to compare and illuminate different aspects of your research problem.

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Once you have developed your problem statement and research questions , you should be ready to choose the specific case that you want to focus on. A good case study should have the potential to:

• Provide new or unexpected insights into the subject
• Challenge or complicate existing assumptions and theories
• Propose practical courses of action to resolve a problem
• Open up new directions for future research

TipIf your research is more practical in nature and aims to simultaneously investigate an issue as you solve it, consider conducting action research instead.

Unlike quantitative or experimental research , a strong case study does not require a random or representative sample. In fact, case studies often deliberately focus on unusual, neglected, or outlying cases which may shed new light on the research problem.

Example of an outlying case studyIn the 1960s the town of Roseto, Pennsylvania was discovered to have extremely low rates of heart disease compared to the US average. It became an important case study for understanding previously neglected causes of heart disease.

However, you can also choose a more common or representative case to exemplify a particular category, experience or phenomenon.

Example of a representative case studyIn the 1920s, two sociologists used Muncie, Indiana as a case study of a typical American city that supposedly exemplified the changing culture of the US at the time.

While case studies focus more on concrete details than general theories, they should usually have some connection with theory in the field. This way the case study is not just an isolated description, but is integrated into existing knowledge about the topic. It might aim to:

• Exemplify a theory by showing how it explains the case under investigation
• Expand on a theory by uncovering new concepts and ideas that need to be incorporated
• Challenge a theory by exploring an outlier case that doesn’t fit with established assumptions

To ensure that your analysis of the case has a solid academic grounding, you should conduct a literature review of sources related to the topic and develop a theoretical framework . This means identifying key concepts and theories to guide your analysis and interpretation.

There are many different research methods you can use to collect data on your subject. Case studies tend to focus on qualitative data using methods such as interviews , observations , and analysis of primary and secondary sources (e.g., newspaper articles, photographs, official records). Sometimes a case study will also collect quantitative data.

Example of a mixed methods case studyFor a case study of a wind farm development in a rural area, you could collect quantitative data on employment rates and business revenue, collect qualitative data on local people’s perceptions and experiences, and analyze local and national media coverage of the development.

The aim is to gain as thorough an understanding as possible of the case and its context.

In writing up the case study, you need to bring together all the relevant aspects to give as complete a picture as possible of the subject.

How you report your findings depends on the type of research you are doing. Some case studies are structured like a standard scientific paper or thesis , with separate sections or chapters for the methods , results and discussion .

Others are written in a more narrative style, aiming to explore the case from various angles and analyze its meanings and implications (for example, by using textual analysis or discourse analysis ).

In all cases, though, make sure to give contextual details about the case, connect it back to the literature and theory, and discuss how it fits into wider patterns or debates.

If you want to know more about statistics , methodology , or research bias , make sure to check out some of our other articles with explanations and examples.

• Normal distribution
• Degrees of freedom
• Null hypothesis
• Discourse analysis
• Control groups
• Mixed methods research
• Non-probability sampling
• Quantitative research
• Ecological validity

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• Rosenthal effect
• Implicit bias
• Cognitive bias
• Selection bias
• Negativity bias
• Status quo bias

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How to Write a Multiple Case Study Effectively

Table of Contents

Have you ever been assigned to write a multiple case study but don’t know where to begin? Are you intimidated by the complexity and challenge it brings? Don’t worry! This article will help you learn how to write a multiple case study effectively that will make an impactful impression. So, let’s begin by defining a multiple case study.

What Is a Multiple Case Study?

A multiple case study is a research method examining several different entities. It helps researchers gain an understanding of the entities’ individual characteristics and disclose any shared patterns or insights. This type of investigation often uses both qualitative and quantitative data. These are usually collected from interviews, surveys, field observations, archival records, and other sources. This is done to analyze the relationships between each entity and its environment. The results can provide valuable insights for policymakers and decision-makers.

Why Is a Multiple Case Study Important?

A multiple case study is invaluable in providing a comprehensive view of a particular issue or phenomenon. Analyzing a range of cases allows for comparisons and contrasts to be drawn. And this can help identify broader trends, implications, and causes that might otherwise remain undetected. This method is particularly useful in developing theories and testing hypotheses. This is because the range of data collected provides more robust evidence than what could be achieved from one single case alone.

How to Write a Multiple Case Study

Below are the key steps on how to write a multiple case study :

1. Brainstorm Potential Case Studies

Before beginning your multiple case study, you should brainstorm potential cases suitable for the research project. Consider both theoretical and practical implications when deciding which cases are most appropriate. Think about how these cases can best illustrate the issue or question at hand. Make sure to consider all relevant information before making any decisions.

2. Conduct Background Research on Each Case

After selecting the individual cases for your multiple case study, the next step is to do background research for each case. Conducting extensive background research on each case will help you better understand the context of the study. This research will allow you to form an educated opinion and provide insight into the problems and challenges that each case may present.

3. Establish a Research Methodology

A successful multiple-case study requires a sound research methodology. This includes deciding on the methods of data collection and analysis and setting objectives. It also involves developing criteria for evaluating the results and determining what kind of data needs to be collected from each case. All of this must be done carefully, considering the purpose of the study and its outcomes.

4. Collect Data

Once a research method has been established, it is time to collect data from each case included in the study. Depending on the nature of the research project, this could involve interviewing participants, gathering statistics, or observing behaviors in certain settings. It is crucial to ensure that all data collected is accurate and reliable.

5. Analyze & Interpret Data

After the data has been collected, it must be analyzed to draw meaningful conclusions from it. This process involves examining patterns and trends within the data, identifying relationships between variables, and looking for commonalities among different cases. These findings must then be interpreted in light of the initial questions posed by the study.

6. Write the Report

After completing the analysis and interpretation of the data, it is finally time to write up the results of the multiple case study. This should include a summary of the key findings and an explanation of why these findings are significant. In addition, the limitations of the study should be acknowledged, along with recommendations for future research in this area.

Writing a multiple case study requires careful planning and execution. But the process becomes easier when you know the proper steps to conduct and create a multiple case study. It requires you to focus on the design of the study, including the sample chosen and the research methodology established. Conducting background research on each case and collecting data are also crucial steps in the process. To guide you through the process, this article outlines the key steps to help you easily write a well-structured multiple-case study .

Abir Ghenaiet

Abir is a data analyst and researcher. Among her interests are artificial intelligence, machine learning, and natural language processing. As a humanitarian and educator, she actively supports women in tech and promotes diversity.

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Continuing to enhance the quality of case study methodology in health services research

Shannon l. sibbald.

1 Faculty of Health Sciences, Western University, London, Ontario, Canada.

2 Department of Family Medicine, Schulich School of Medicine and Dentistry, Western University, London, Ontario, Canada.

3 The Schulich Interfaculty Program in Public Health, Schulich School of Medicine and Dentistry, Western University, London, Ontario, Canada.

Stefan Paciocco

Meghan fournie, rachelle van asseldonk, tiffany scurr.

Case study methodology has grown in popularity within Health Services Research (HSR). However, its use and merit as a methodology are frequently criticized due to its flexible approach and inconsistent application. Nevertheless, case study methodology is well suited to HSR because it can track and examine complex relationships, contexts, and systems as they evolve. Applied appropriately, it can help generate information on how multiple forms of knowledge come together to inform decision-making within healthcare contexts. In this article, we aim to demystify case study methodology by outlining its philosophical underpinnings and three foundational approaches. We provide literature-based guidance to decision-makers, policy-makers, and health leaders on how to engage in and critically appraise case study design. We advocate that researchers work in collaboration with health leaders to detail their research process with an aim of strengthening the validity and integrity of case study for its continued and advanced use in HSR.

Introduction

The popularity of case study research methodology in Health Services Research (HSR) has grown over the past 40 years. 1 This may be attributed to a shift towards the use of implementation research and a newfound appreciation of contextual factors affecting the uptake of evidence-based interventions within diverse settings. 2 Incorporating context-specific information on the delivery and implementation of programs can increase the likelihood of success. 3 , 4 Case study methodology is particularly well suited for implementation research in health services because it can provide insight into the nuances of diverse contexts. 5 , 6 In 1999, Yin 7 published a paper on how to enhance the quality of case study in HSR, which was foundational for the emergence of case study in this field. Yin 7 maintains case study is an appropriate methodology in HSR because health systems are constantly evolving, and the multiple affiliations and diverse motivations are difficult to track and understand with traditional linear methodologies.

Despite its increased popularity, there is debate whether a case study is a methodology (ie, a principle or process that guides research) or a method (ie, a tool to answer research questions). Some criticize case study for its high level of flexibility, perceiving it as less rigorous, and maintain that it generates inadequate results. 8 Others have noted issues with quality and consistency in how case studies are conducted and reported. 9 Reporting is often varied and inconsistent, using a mix of approaches such as case reports, case findings, and/or case study. Authors sometimes use incongruent methods of data collection and analysis or use the case study as a default when other methodologies do not fit. 9 , 10 Despite these criticisms, case study methodology is becoming more common as a viable approach for HSR. 11 An abundance of articles and textbooks are available to guide researchers through case study research, including field-specific resources for business, 12 , 13 nursing, 14 and family medicine. 15 However, there remains confusion and a lack of clarity on the key tenets of case study methodology.

Several common philosophical underpinnings have contributed to the development of case study research 1 which has led to different approaches to planning, data collection, and analysis. This presents challenges in assessing quality and rigour for researchers conducting case studies and stakeholders reading results.

This article discusses the various approaches and philosophical underpinnings to case study methodology. Our goal is to explain it in a way that provides guidance for decision-makers, policy-makers, and health leaders on how to understand, critically appraise, and engage in case study research and design, as such guidance is largely absent in the literature. This article is by no means exhaustive or authoritative. Instead, we aim to provide guidance and encourage dialogue around case study methodology, facilitating critical thinking around the variety of approaches and ways quality and rigour can be bolstered for its use within HSR.

Purpose of case study methodology

Case study methodology is often used to develop an in-depth, holistic understanding of a specific phenomenon within a specified context. 11 It focuses on studying one or multiple cases over time and uses an in-depth analysis of multiple information sources. 16 , 17 It is ideal for situations including, but not limited to, exploring under-researched and real-life phenomena, 18 especially when the contexts are complex and the researcher has little control over the phenomena. 19 , 20 Case studies can be useful when researchers want to understand how interventions are implemented in different contexts, and how context shapes the phenomenon of interest.

In addition to demonstrating coherency with the type of questions case study is suited to answer, there are four key tenets to case study methodologies: (1) be transparent in the paradigmatic and theoretical perspectives influencing study design; (2) clearly define the case and phenomenon of interest; (3) clearly define and justify the type of case study design; and (4) use multiple data collection sources and analysis methods to present the findings in ways that are consistent with the methodology and the study’s paradigmatic base. 9 , 16 The goal is to appropriately match the methods to empirical questions and issues and not to universally advocate any single approach for all problems. 21

Approaches to case study methodology

Three authors propose distinct foundational approaches to case study methodology positioned within different paradigms: Yin, 19 , 22 Stake, 5 , 23 and Merriam 24 , 25 ( Table 1 ). Yin is strongly post-positivist whereas Stake and Merriam are grounded in a constructivist paradigm. Researchers should locate their research within a paradigm that explains the philosophies guiding their research 26 and adhere to the underlying paradigmatic assumptions and key tenets of the appropriate author’s methodology. This will enhance the consistency and coherency of the methods and findings. However, researchers often do not report their paradigmatic position, nor do they adhere to one approach. 9 Although deliberately blending methodologies may be defensible and methodologically appropriate, more often it is done in an ad hoc and haphazard way, without consideration for limitations.

Cross-analysis of three case study approaches, adapted from Yazan 2015

The post-positive paradigm postulates there is one reality that can be objectively described and understood by “bracketing” oneself from the research to remove prejudice or bias. 27 Yin focuses on general explanation and prediction, emphasizing the formulation of propositions, akin to hypothesis testing. This approach is best suited for structured and objective data collection 9 , 11 and is often used for mixed-method studies.

Constructivism assumes that the phenomenon of interest is constructed and influenced by local contexts, including the interaction between researchers, individuals, and their environment. 27 It acknowledges multiple interpretations of reality 24 constructed within the context by the researcher and participants which are unlikely to be replicated, should either change. 5 , 20 Stake and Merriam’s constructivist approaches emphasize a story-like rendering of a problem and an iterative process of constructing the case study. 7 This stance values researcher reflexivity and transparency, 28 acknowledging how researchers’ experiences and disciplinary lenses influence their assumptions and beliefs about the nature of the phenomenon and development of the findings.

Defining a case

A key tenet of case study methodology often underemphasized in literature is the importance of defining the case and phenomenon. Researches should clearly describe the case with sufficient detail to allow readers to fully understand the setting and context and determine applicability. Trying to answer a question that is too broad often leads to an unclear definition of the case and phenomenon. 20 Cases should therefore be bound by time and place to ensure rigor and feasibility. 6

Yin 22 defines a case as “a contemporary phenomenon within its real-life context,” (p13) which may contain a single unit of analysis, including individuals, programs, corporations, or clinics 29 (holistic), or be broken into sub-units of analysis, such as projects, meetings, roles, or locations within the case (embedded). 30 Merriam 24 and Stake 5 similarly define a case as a single unit studied within a bounded system. Stake 5 , 23 suggests bounding cases by contexts and experiences where the phenomenon of interest can be a program, process, or experience. However, the line between the case and phenomenon can become muddy. For guidance, Stake 5 , 23 describes the case as the noun or entity and the phenomenon of interest as the verb, functioning, or activity of the case.

Designing the case study approach

Yin’s approach to a case study is rooted in a formal proposition or theory which guides the case and is used to test the outcome. 1 Stake 5 advocates for a flexible design and explicitly states that data collection and analysis may commence at any point. Merriam’s 24 approach blends both Yin and Stake’s, allowing the necessary flexibility in data collection and analysis to meet the needs.

Yin 30 proposed three types of case study approaches—descriptive, explanatory, and exploratory. Each can be designed around single or multiple cases, creating six basic case study methodologies. Descriptive studies provide a rich description of the phenomenon within its context, which can be helpful in developing theories. To test a theory or determine cause and effect relationships, researchers can use an explanatory design. An exploratory model is typically used in the pilot-test phase to develop propositions (eg, Sibbald et al. 31 used this approach to explore interprofessional network complexity). Despite having distinct characteristics, the boundaries between case study types are flexible with significant overlap. 30 Each has five key components: (1) research question; (2) proposition; (3) unit of analysis; (4) logical linking that connects the theory with proposition; and (5) criteria for analyzing findings.

Contrary to Yin, Stake 5 believes the research process cannot be planned in its entirety because research evolves as it is performed. Consequently, researchers can adjust the design of their methods even after data collection has begun. Stake 5 classifies case studies into three categories: intrinsic, instrumental, and collective/multiple. Intrinsic case studies focus on gaining a better understanding of the case. These are often undertaken when the researcher has an interest in a specific case. Instrumental case study is used when the case itself is not of the utmost importance, and the issue or phenomenon (ie, the research question) being explored becomes the focus instead (eg, Paciocco 32 used an instrumental case study to evaluate the implementation of a chronic disease management program). 5 Collective designs are rooted in an instrumental case study and include multiple cases to gain an in-depth understanding of the complexity and particularity of a phenomenon across diverse contexts. 5 , 23 In collective designs, studying similarities and differences between the cases allows the phenomenon to be understood more intimately (for examples of this in the field, see van Zelm et al. 33 and Burrows et al. 34 In addition, Sibbald et al. 35 present an example where a cross-case analysis method is used to compare instrumental cases).

Merriam’s approach is flexible (similar to Stake) as well as stepwise and linear (similar to Yin). She advocates for conducting a literature review before designing the study to better understand the theoretical underpinnings. 24 , 25 Unlike Stake or Yin, Merriam proposes a step-by-step guide for researchers to design a case study. These steps include performing a literature review, creating a theoretical framework, identifying the problem, creating and refining the research question(s), and selecting a study sample that fits the question(s). 24 , 25 , 36

Data collection and analysis

Using multiple data collection methods is a key characteristic of all case study methodology; it enhances the credibility of the findings by allowing different facets and views of the phenomenon to be explored. 23 Common methods include interviews, focus groups, observation, and document analysis. 5 , 37 By seeking patterns within and across data sources, a thick description of the case can be generated to support a greater understanding and interpretation of the whole phenomenon. 5 , 17 , 20 , 23 This technique is called triangulation and is used to explore cases with greater accuracy. 5 Although Stake 5 maintains case study is most often used in qualitative research, Yin 17 supports a mix of both quantitative and qualitative methods to triangulate data. This deliberate convergence of data sources (or mixed methods) allows researchers to find greater depth in their analysis and develop converging lines of inquiry. For example, case studies evaluating interventions commonly use qualitative interviews to describe the implementation process, barriers, and facilitators paired with a quantitative survey of comparative outcomes and effectiveness. 33 , 38 , 39

Yin 30 describes analysis as dependent on the chosen approach, whether it be (1) deductive and rely on theoretical propositions; (2) inductive and analyze data from the “ground up”; (3) organized to create a case description; or (4) used to examine plausible rival explanations. According to Yin’s 40 approach to descriptive case studies, carefully considering theory development is an important part of study design. “Theory” refers to field-relevant propositions, commonly agreed upon assumptions, or fully developed theories. 40 Stake 5 advocates for using the researcher’s intuition and impression to guide analysis through a categorical aggregation and direct interpretation. Merriam 24 uses six different methods to guide the “process of making meaning” (p178) : (1) ethnographic analysis; (2) narrative analysis; (3) phenomenological analysis; (4) constant comparative method; (5) content analysis; and (6) analytic induction.

Drawing upon a theoretical or conceptual framework to inform analysis improves the quality of case study and avoids the risk of description without meaning. 18 Using Stake’s 5 approach, researchers rely on protocols and previous knowledge to help make sense of new ideas; theory can guide the research and assist researchers in understanding how new information fits into existing knowledge.

Practical applications of case study research

Columbia University has recently demonstrated how case studies can help train future health leaders. 41 Case studies encompass components of systems thinking—considering connections and interactions between components of a system, alongside the implications and consequences of those relationships—to equip health leaders with tools to tackle global health issues. 41 Greenwood 42 evaluated Indigenous peoples’ relationship with the healthcare system in British Columbia and used a case study to challenge and educate health leaders across the country to enhance culturally sensitive health service environments.

An important but often omitted step in case study research is an assessment of quality and rigour. We recommend using a framework or set of criteria to assess the rigour of the qualitative research. Suitable resources include Caelli et al., 43 Houghten et al., 44 Ravenek and Rudman, 45 and Tracy. 46

New directions in case study

Although “pragmatic” case studies (ie, utilizing practical and applicable methods) have existed within psychotherapy for some time, 47 , 48 only recently has the applicability of pragmatism as an underlying paradigmatic perspective been considered in HSR. 49 This is marked by uptake of pragmatism in Randomized Control Trials, recognizing that “gold standard” testing conditions do not reflect the reality of clinical settings 50 , 51 nor do a handful of epistemologically guided methodologies suit every research inquiry.

Pragmatism positions the research question as the basis for methodological choices, rather than a theory or epistemology, allowing researchers to pursue the most practical approach to understanding a problem or discovering an actionable solution. 52 Mixed methods are commonly used to create a deeper understanding of the case through converging qualitative and quantitative data. 52 Pragmatic case study is suited to HSR because its flexibility throughout the research process accommodates complexity, ever-changing systems, and disruptions to research plans. 49 , 50 Much like case study, pragmatism has been criticized for its flexibility and use when other approaches are seemingly ill-fit. 53 , 54 Similarly, authors argue that this results from a lack of investigation and proper application rather than a reflection of validity, legitimizing the need for more exploration and conversation among researchers and practitioners. 55

Although occasionally misunderstood as a less rigourous research methodology, 8 case study research is highly flexible and allows for contextual nuances. 5 , 6 Its use is valuable when the researcher desires a thorough understanding of a phenomenon or case bound by context. 11 If needed, multiple similar cases can be studied simultaneously, or one case within another. 16 , 17 There are currently three main approaches to case study, 5 , 17 , 24 each with their own definitions of a case, ontological and epistemological paradigms, methodologies, and data collection and analysis procedures. 37

Individuals’ experiences within health systems are influenced heavily by contextual factors, participant experience, and intricate relationships between different organizations and actors. 55 Case study research is well suited for HSR because it can track and examine these complex relationships and systems as they evolve over time. 6 , 7 It is important that researchers and health leaders using this methodology understand its key tenets and how to conduct a proper case study. Although there are many examples of case study in action, they are often under-reported and, when reported, not rigorously conducted. 9 Thus, decision-makers and health leaders should use these examples with caution. The proper reporting of case studies is necessary to bolster their credibility in HSR literature and provide readers sufficient information to critically assess the methodology. We also call on health leaders who frequently use case studies 56 – 58 to report them in the primary research literature.

The purpose of this article is to advocate for the continued and advanced use of case study in HSR and to provide literature-based guidance for decision-makers, policy-makers, and health leaders on how to engage in, read, and interpret findings from case study research. As health systems progress and evolve, the application of case study research will continue to increase as researchers and health leaders aim to capture the inherent complexities, nuances, and contextual factors. 7

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A multiple case study is a research method that uses multiple research sites to gain a more comprehensive understanding of a particular phenomenon. It examines multiple cases in order to analyze patterns and the relationships between variables. This type of research method is used in management to gain a deeper understanding of a particular problem or issue. It is a systematic approach to gathering and analyzing data from multiple different sources such as individuals, organizations, or communities. The multiple case study approach allows researchers to gain greater insight into complex problems by considering a variety of perspectives, contexts, and sources of information .

• 1 Example of multiple case study
• 2 When to use multiple case study
• 3 Types of multiple case study
• 4 Steps of multiple case study
• 5 Limitations of multiple case study
• 6 Other approaches related to multiple case study
• 7 References

Example of multiple case study

• A multiple case study example could be a study of different companies in the same industry in order to analyze the differences in their strategies and performance. For instance, a researcher may examine three companies in the automotive industry and determine what strategies have been successful and which have not. They may then compare the results of these three companies in order to determine which strategies are most effective.
• Another example of a multiple case study could be an examination of how different countries have responded to the COVID-19 pandemic. In this case, the researcher could look at different strategies adopted by countries worldwide and analyze the results of those strategies. They could then compare the results in order to determine which strategies have been most successful in mitigating the spread of the virus.
• A third example could be a study of different schools and how they have adapted to the online learning environment . The researcher could look at the successes and failures of different schools in order to determine which strategies are most effective in transitioning to remote learning. They could then use these findings to suggest changes and improvements to the schools’ policies and procedures .

When to use multiple case study

A multiple case study approach is a useful tool for researchers looking to gain a deeper understanding of complex issues. This method can be used in a variety of contexts, such as studying organizational management , social phenomena, or public health interventions. It can provide a more comprehensive understanding of the problem by considering a variety of perspectives, contexts, and sources of information. Examples of when multiple case studies can be used include:

• Examining the effectiveness of a particular policy or program in multiple contexts.
• Exploring the dynamics of organizational change across different settings.
• Investigating the impact of a cultural or social phenomenon on different communities.
• Analyzing the differences in responses to a public health intervention between populations.
• Understanding the dynamics of an issue in order to inform the development of new policies or practices.

Types of multiple case study

• Exploratory multiple case study: An exploratory multiple case study is used to explore a research problem in greater detail. It is used when the research question or problem is not well-defined, or when the researcher is uncertain about the best approach to study the problem. This type of multiple case study is often used to generate new ideas and to identify potential research topics.
• Explanatory Multiple Case Study: An explanatory multiple case study is used to explain a research problem in detail. It is used when the researcher is looking to explain the cause of an event or phenomenon. This type of multiple case study is used to identify patterns and relationships between variables, and to identify potential explanations for the phenomenon being studied.
• Descriptive Multiple Case Study: A descriptive multiple case study is used to describe a research problem in detail. It is used when the researcher wants to provide a comprehensive overview of a particular topic or phenomenon. This type of multiple case study is useful for providing a detailed description of a particular event or phenomenon and its context.
• Comparative Multiple Case Study: A comparative multiple case study is used to compare two or more research sites. It is used when the researcher wants to compare and contrast a phenomenon across multiple sites. This type of multiple case study is useful for examining similarities and differences between different research sites.
• Embedded Multiple Case Study: An embedded multiple case study is used to embed a single case study within a larger research project . It is used when the researcher wants to incorporate a single case study within a larger research project. This type of multiple case study is useful for exploring the complexities of a particular research problem, and for providing an in-depth understanding of a particular phenomenon.

Steps of multiple case study

A multiple case study is a research method that uses multiple research sites to gain a more comprehensive understanding of a particular phenomenon. The multiple case study approach allows researchers to gain greater insight into complex problems by considering a variety of perspectives, contexts, and sources of information. The following steps are necessary for conducting a successful multiple case study:

• Selecting the research sites : The first step in a multiple case study is to select the research sites. This requires careful consideration of factors such as the size and scope of the problem, the availability of data and resources, and the accessibility of the research sites.
• Gathering data : After selecting the research sites, the next step is to gather data. This can be done through interviews, surveys, focus groups, and other data collection methods .
• Analyzing the data : Once the data has been gathered, it must be analyzed in order to identify patterns and relationships between variables. This requires careful analysis of the data and may involve using statistical methods such as regression and factor analysis.
• Drawing conclusions : After the data has been analyzed, the next step is to draw conclusions. This involves synthesizing the data and making sense of it in order to answer the research question.
• Reporting the results : The final step is to report the results of the multiple case study. This can be done through a written report, a presentation, or a multimedia format.

Limitations of multiple case study

Multiple case studies have some limitations that should be taken into consideration when using this method. These limitations include:

• The multiple case study approach can be time consuming and resource intensive, as researchers must collect and analyze data from multiple different sources.
• It can be difficult to identify patterns and relationships between variables when studying multiple cases.
• The data collected from multiple cases may be difficult to generalize to a larger population.
• The multiple case study approach is limited to studying phenomena in limited contexts, and does not provide a holistic picture of a phenomenon.
• It can be difficult to control for all variables in a multiple case study, which can lead to inaccurate results.

Other approaches related to multiple case study

A multiple case study is a research method that uses multiple research sites to gain a more comprehensive understanding of a particular phenomenon. Other approaches related to multiple case studies include:

• Qualitative research : Qualitative research is an empirical research approach which focuses on understanding the perspectives, experiences, and beliefs of people in their contexts. It typically involves interviews, observations, and other forms of data collection.
• Grounded Theory : Grounded theory is an inductive research method that examines how social processes are created, maintained, and changed. It involves the systematic collection and analysis of data to generate new theory.
• Action Research : Action research is a type of research that involves the active participation of stakeholders in the research process . It focuses on identifying and resolving practical problems in an organization or community.
• Gustafsson, J. (2017). Single case studies vs. multiple case studies: A comparative study .
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• Published: 02 April 2024

Adopting, implementing and assimilating coproduced health and social care innovations involving structurally vulnerable populations: findings from a longitudinal, multiple case study design in Canada, Scotland and Sweden

• Gillian Mulvale   ORCID: orcid.org/0000-0003-0546-6910 1 ,
• Jenn Green 1 ,
• Glenn Robert 2 , 5 ,
• Michael Larkin 3 ,
• Nicoline Vackerberg 4 , 5 ,
• Sofia Kjellström 5 ,
• Puspita Hossain 6 ,
• Sandra Moll 7 ,
• Esther Lim 8 , 9 &
• Shioma-Lei Craythorne 3  

Health Research Policy and Systems volume  22 , Article number:  42 ( 2024 ) Cite this article

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Innovations in coproduction are shaping public service reform in diverse contexts around the world. Although many innovations are local, others have expanded and evolved over time. We know very little, however, about the process of implementation and evolution of coproduction. The purpose of this study was to explore the adoption, implementation and assimilation of three approaches to the coproduction of public services with structurally vulnerable groups.

We conducted a 4 year longitudinal multiple case study (2019–2023) of three coproduced public service innovations involving vulnerable populations: ESTHER in Jönköping Region, Sweden involving people with multiple complex needs (Case 1); Making Recovery Real in Dundee, Scotland with people who have serious mental illness (Case 2); and Learning Centres in Manitoba, Canada (Case 3), also involving people with serious mental illness. Data sources included 14 interviews with strategic decision-makers and a document analysis to understand the history and contextual factors relating to each case. Three frameworks informed the case study protocol, semi-structured interview guides, data extraction, deductive coding and analysis: the Consolidated Framework for Implementation Research, the Diffusion of Innovation model and Lozeau’s Compatibility Gaps to understand assimilation.

The adoption of coproduction involving structurally vulnerable populations was a notable evolution of existing improvement efforts in Cases 1 and 3, while impetus by an external change agency, existing collaborative efforts among community organizations, and the opportunity to inform a new municipal mental health policy sparked adoption in Case 2. In all cases, coproduced innovation centred around a central philosophy that valued lived experience on an equal basis with professional knowledge in coproduction processes. This philosophical orientation offered flexibility and adaptability to local contexts, thereby facilitating implementation when compared with more defined programming. According to the informants, efforts to avoid co-optation risks were successful, resulting in the assimilation of new mindsets and coproduction processes, with examples of how this had led to transformative change.

Conclusions

In exploring innovations in coproduction with structurally vulnerable groups, our findings suggest several additional considerations when applying existing theoretical frameworks. These include the philosophical nature of the innovation, the need to study the evolution of the innovation itself as it emerges over time, greater attention to partnered processes as disruptors to existing power structures and an emphasis on driving transformational change in organizational cultures.

Peer Review reports

Growing recognition by governments internationally of the need to involve the perspectives of people using public services when designing, delivering and improving those services has been described as a Participatory Zeitgeist reflecting the “spirit of our time” [ 1 , 2 (p247)]. Researchers and designers have developed various approaches drawn from different disciplines and using different labels (for example, codesign, cocreation, coproduction) that align with principles in the citizen engagement literature [ 3 ]. These approaches recognize that service users have experiences and assets and can contribute to service design and delivery along with professional expertise, rather than simply being passive recipients of services designed and delivered by others [ 3 , 4 ]. While these approaches can be used with anyone, they have been increasingly applied to promote the inclusion of structurally vulnerable populations in the design and delivery of innovative health and social care services that seek to support them.

While coproduction has the potential to reform inequitable structures and social processes, excluding vulnerable groups or involving them in a tokenistic manner may unintentionally reinforce existing power imbalances [ 4 , 5 ]. For example, gaps have been noted between the rhetoric of service user involvement in international mental health policy and the readiness to adopt such policies in practice [ 6 ]. Challenges have also been noted in incorporating the voices of individuals with complex needs in improving care coordination across health and social services [ 7 ].

Despite increasing attention to coproduction in the literature and practice, knowledge gaps exist with respect to the implementation of coproduction involving vulnerable populations in different contexts [ 8 , 9 , 10 ]. An international symposium of coproduction researchers and people with lived experience held in Birmingham, England in 2017 identified the need for research to understand how exemplary coproduction innovations involving structurally vulnerable groups originated and their assimilation into routine practice [ 11 ]. To our knowledge, established implementation science models have yet to be applied to coproduction, where service users and service providers are cocreating innovations during the process of implementation [ 12 ].

In this paper, we present findings from a longitudinal case study exploring the factors and processes that influence the adoption, implementation and assimilation of three diverse coproduced public service innovations involving structurally vulnerable groups. We explored the perspectives of strategic leaders involved in advancing coproduction processes involving vulnerable groups. Our analysis proceeds through the lens of existing frameworks from the literature to discuss the outer context (economic, social, political, geographical), inner context (organizational and community considerations), individual factors, innovation features and process considerations [ 13 ].

Conceptual foundations: coproduction, structural vulnerability and implementation processes

Coproduction: Coproduction has been defined as “… involvement of public service users in the design, management, delivery and/or evaluation of public services” [ 4 ]. A core feature of coproduction approaches is that they are applied in a flexible manner, dynamically and innovatively responding to local needs and context [ 14 ].

Structural vulnerability: We adopt the term structurally vulnerable populations to recognize that vulnerability is not inherent in these populations but rather in the social, economic and political systems in which they are embedded [ 15 , 16 ]. Examples include individuals who may require multiple health and/or other public services, including people with complex and intersecting health needs (for example, heart failure and dementia) along with poverty, homelessness and/or being members of newcomer or racialized groups. Structural barriers (for example, lack of trust, language, cultural, scheduling, financial) and power relations may prevent them from engaging in coproduction.

Adoption, implementation and assimilation: We draw on and combine elements from three theoretical frameworks to guide this research. The first is the Diffusion Of Innovation (DOI) model [ 17 ], which identifies how political, social, economic, cultural, and organizational factors and processes affect fidelity and adoption during the diffusion of service innovation. The second is the Consolidated Framework for Implementation Research (CFIR) [ 18 , 19 ], which demonstrates the importance of contextual factors at multiple levels (external context, internal context, innovation features, processes and individual characteristics) in shaping the implementation of service improvements. The third is Lozeau et al.’s (2002) compatibility gaps [ 20 ], which characterize different forms of assimilation of innovations into routine practice [ 20 , 21 ]. Based on these frameworks, we define innovation as a novel set of behaviours, routines and ways of working that are directed at improving health outcomes, administrative efficiency, cost effectiveness or users’ experiences, and that are implemented by planned and coordinated action [ 20 ]. We define adoption as the incremental considerations and progressive individual and collective decision-making from pre-contemplation through exploration by which organizations ultimately decide to adopt the innovation (programme/model/process). Implementation describes the formal strategies to promote the integration of innovations into existing practices. Assimilation is the informal process by which, over time, innovations become part of routine ways of doing things. Assimilation can be characterized as (a) transformation when there is high fidelity to the model and the organization adjusts its functioning to fit the assumptions of the model; (b) customization when the model is adapted to the context and the organization adjusts its practices; (c) loose coupling whereby the innovation is adopted only superficially, while the functioning of the organization remains largely unaffected; or (d) co-optation whereby the innovation becomes captured and distorted to reinforce existing organizational roles and power structures [ 21 ].

Study aim and design

We adopt a longitudinal multiple case study approach to understand the dynamic nature by which three coproduced innovations intended to address the needs of vulnerable populations were adopted, implemented and assimilated [ 22 ]. Case study research is well suited to studying contemporary phenomena in their real-life contexts, and theory is often adopted to focus the analysis, allowing the theory to be augmented or revised based on emerging findings [ 22 ]. To meet the criteria of being a ‘case’, an innovation had to be underpinned by a coproduction model involving structurally vulnerable populations in the design, management, delivery and/or evaluation of a public service that has advanced through these phases. Concepts from the CFIR, DOI and assimilation frameworks described above informed the case study protocol, semi-structured interview questions, data extraction and coding.

Case selection

The three cases were selected through the networks of the investigators to illustrate how coproduction involving vulnerable populations can be advanced in different contexts: the region of Jönköping, Sweden striving for better patient outcomes and experiences by tailoring care to the needs of people with multiple complex needs (Case 1 – ESTHER); the city of Dundee, Scotland aiming to advance the recovery of people with mental illness through greater collaboration with those with lived experience and among service organizations (Case 2 – Making Recovery Real [MRR]); and a rural and an urban branch of a national community mental health organization in a Canadian province that adapted the English Recovery College model of coproduced educational programming to support the recovery of people with serious mental illness (Case 3 – Canadian Mental Health Association [CMHA] Manitoba and Winnipeg and CMHA Central branches’ Learning Centres in Manitoba, Canada) (see Tables  1 , 2 and 3 ).

The study team were familiar with each of these cases and were confident in having good access to them over time. Additionally, their different national contexts offered the opportunity to consider macro-level factors. While each of these countries’ health and social care systems are largely publicly funded, funding is the responsibility of different levels of government (municipal, provincial and/or national) and services are administered and delivered primarily by local governments and/or designated authorities (see Table  4 ).

Data sources and collection

Data sources include relevant academic and grey literature identified through electronic searches and/or recommended or shared by local gatekeepers and key informants to inform the background case context for the individual case analyses, and the interview guides (see Table  5 , and Table S1 in Additional file 1 for more details). Research team members (GM, JG, GR, NV, PH, SC, SS) conducted 45–60 minute long semi-structured interviews in person or online between November 2019 and August 2021. To help understand the history and context of each case, key informants were strategic decision-makers and programme managers affiliated at the time with the organizations leading, participating in or supporting the local initiatives, and who were familiar with the history of how the coproduced innovations emerged, their developmental timeline and coproduction’s role in the overall system. Footnote 1

The interview guide questions probed about this history with a focus on the contextual factors that influenced adoption and implementation and the extent to which coproduction has been assimilated into routine practice. Data were gathered through investigator field notes, the audio-recording and transcription of interviews, timelines, hand-written notes and/or audio-recordings of team meetings to capture member checking with local collaborators, and case team memos of decision points.

To maintain participant anonymity, participant codes are used in the text, identified by a location code (for Case 1, JKG = Jönköping, Sweden; for Case 2, DND = Dundee, Scotland; for Case 3, OTH = Other [for example, national, international informants], PLP = Portage la Prairie, Manitoba, Canada; WPG = Winnipeg, Manitoba, Canada), and a participant number (that is, 01, 02, 03 and so on). For example, an informant from Dundee could be DND-03. Note that the perspectives of service providers and people with lived experience of structural vulnerability were not the focus here but are considered in subsequent waves of our data collection to understand their experiences of coproduction in practice.

Data analysis

A common coding framework was developed iteratively to capture factors and processes influencing adoption, implementation and assimilation by combining elements of the theoretical frameworks to remove overlap and promote consistency of understanding when coding and interpreting the data. Table S2 presents this in more detail (see Additional file 2 ).

The initial data extraction was performed by the research team affiliated with each case, and the project research coordinator worked with the local research coordinator for each case to ensure consistency across cases. Documentary evidence analysis primarily informed our understanding of the historical context and overview of each case. All data were coded and analysed using a deductive approach; a common coding scheme and thematic analysis were employed, respectively, based on the theoretical propositions and concepts in the CFIR and DOI models, and allowing for emergent themes, particularly in relation to the coproduction context [ 22 ]. A visual timeline was created to understand the initiation and growth of coproduction in each case. Interview data was triangulated with documentary evidence and field notes. Analysis proceeded on a case-by-case basis, followed by a cross-case analysis.

Qualitative validity and reliability

The research team comprised four members who were familiar with one of the three cases prior to the study (the ESTHER case), as well as eight members who were not familiar with any of the cases. One member of the team had been closely involved with the development of the ESTHER case over a long period of time. The use of a common and detailed case study protocol and data management system, central and local research coordination by case, monthly investigator meetings and tri-annual full team meetings including collaborating organization representatives were strategies used to enhance qualitative validity. The common coding framework and frequent team discussions helped to ensure consistency and enhanced reliability. Data were triangulated across sources, the analysis was triangulated across investigators and theories, and member checked at various stages with the full team of investigators and collaborators [ 23 ].

Ethical considerations

Research ethics clearance was obtained from the relevant academic research ethics boards (McMaster University Research Ethics Board [MREB Project ID 2066], Aston University Ethics Committee [Rec Ref #1611]; King’s College London Research Ethics Office [Reference Number MOD-19/20-17350]; SingHealth Centralized Institutional Review Board [CIRB Ref# 2020/2341]; and Swedish Ethical Review Authority [Etikprövningsmyndigheten, Dnr 2019-06373]), and in light of this, ethics review was waived by the boards of the collaborating organizations (Canadian Mental Health Association, Manitoba & Winnipeg branch, the East of Scotland Research Ethics Service). Participants received letters of information outlining the study objectives, protocol and risks prior to consenting in writing. Data were collected and stored locally and shared across sites as anonymized, encrypted and password-protected files.

We outline the historical context and analysis of contextual factors influencing adoption and implementation, discuss assimilation by case and then present a cross-case analysis. Tables 1 , 2 and 3 above capture the key features of each case, Figs.  1 , 2 and 3 summarize the adoption, implementation and assimilation timelines, and Tables  6 , 7 and 8 summarize the cross-case analysis.

Historical context: ESTHER is a complex system of public health and social care services run by 13 municipal councils in Region Jönköping County, Sweden that has brought intersectoral health and social care providers together since the 1990s to increase coordination and to redefine service experiences around the needs of the person receiving the services. In a context of restricted public sector funding, ESTHER began in 1997, initially for 2 years, with the aim of finding ways to meet population health needs using approaches other than increased hospital bed capacity. Hospital leaders in Region Jönköping County aimed to transform ways of working and to prevent hospital admissions through what informants called “radical customization”, which considered the needs of individual patients using a bottom-up change process referred to as health process re-engineering. This approach 'shadowed' a patient with complex needs through their health service experience journey and included interviews and surveys with patients, staff and government officials and observations of care encounters and processes to gain new insights into what was needed to improve the system from the patient perspectives. Storytelling of the experience of 'Esther', a persona of an elderly person with complex health needs, actualized this process, pointing out what needed to be done differently by demonstrating the importance of focusing on the experience of the person receiving care. The lessons learned from ESTHER fuelled health and social service-wide change, including coproduction with patients beginning in 2006 through patient roles on advisory committees and councils, and has expanded to include initiatives such as ESTHER cafes, ESTHER coach training and ESTHER family meetings, among others.

Adoption: In the ESTHER case 'adoption' of coproduction was an emergent phenomenon that took place over a 10 year period as ongoing improvement efforts, aimed from the outset at better capturing the lived experience of people with complex needs, evolved in terms of how their perspectives were incorporated in design and decision-making. This initially began with interviews and shadowing patients and bringing staff on board with this approach, until by 2006, Esthers became more directly involved in coproducing system improvements. In the internal context , healthcare process re-engineering efforts since the 1990s centred on the question of “What is best for Esther?” and demonstrated the importance of person-centred care and emphasizing the experiences of the person in need of complex care, laying the foundation for a coproduction approach to emerge. In the external context , system-wide efforts by health and social leaders to create a system map led to ESTHER becoming more than a health quality improvement project but rather a health and social systems-wide movement. From a process perspective, the initial project’s evaluation results indicated a 20% reduction in hospital beds, an achievement that earned recognition in the external context through two national awards. As project funding ended, the benefits of the ESTHER philosophy were recognized, and ESTHER transitioned from a project to a 'network' without funding. Over the next few years, the ESTHER Network further developed as 'cousins' emerged across Sweden, and the approach was adopted in other countries, including Italy, England, Scotland and France.

By 2006, ESTHER in Sweden transitioned toward adopting coproduction approaches that actively invited participation of people with lived experience expertise (Esthers) in coproducing ongoing innovations; however, this process was emergent and not uniform. The flexibility of a guiding philosophy was a key feature that enabled this emergence of innovation in the coproduction approach. By this time, some individual system leaders had come to recognize that keeping the focus on value and what is best for the person being treated in their daily lives would lead to better results than a preoccupation with resources and cost cutting. ESTHER had transformed relationships internally in hospitals to team-based (doctor‒nurse) coleadership and externally across the region via interorganizational collaboration between hospitals, primary care, community care and social care to improve Esthers’ care journeys. These collaborative ways of working were preparation for collaboration with Esthers, helping to create receptivity among senior leaders to coproduction. Nonetheless, at this stage of adoption there was still some internal resistance, particularly at middle management and staff levels, as Esthers began attending and sharing stories about their experiences at leadership meetings.

“I think one of the most important decisions was to take patient in the room. In addition, there was a lot of resistance”. [JKG-01]

Implementation: Once the decision to work directly with Esthers was taken, the implementation of coproduction has continued to unfold, albeit unevenly and opportunistically. Around this time, factors in the outer context shaped ESTHER’s continued development, as Esthers became increasingly present in local patient committees and began to participate in and influence the ESTHER steering committee. While ongoing primary care reform was a distraction for many health service managers, an external network of Esthers developed from different programmes across municipalities, and annual ESTHER 'family' meetings were held, where Esthers could convene to share experiences and ideas, strengthening the grassroots support. ESTHER was again gaining international recognition, becoming the subject of a BBC documentary film and being declared “one of the coolest innovations in the world” by CNN.

In the internal context , further developments included the creation of internal structures that were funded to support greater involvement of patients with multiple vulnerabilities in coproduction activities: The ESTHER Competence Center, training healthcare teams to follow the ESTHER philosophy, and ESTHER Coach quality improvement training programmes for approximately 30 health and social service providers to become new ESTHER Coaches each year, and with growing numbers of Esthers as faculty. Key features of the approach were supportive of grassroots growth. Coaches developed innovations on an ongoing basis with input from Esthers, and health and social service providers remarked that the ESTHER philosophy takes them back to the reasons they entered their professions. At the same time, the bottom-up nature driving innovation continued to be threatening to some individuals in senior leadership positions who were more distanced from observing the benefits.

“ESTHER is very much bottom-up. So, you are very close to ESTHER … you see what’s going on and what you can do better. The steering is from the bottom, and then the managers got a bit threatened. I think there was suddenly too much; the movement was suddenly too big. So, people were reacting to that. …That still is a challenge”. [JKG-01]

Creative approaches have been used to foster growth despite this resistance. Small changes such as renaming committees have enabled participation by Esthers.

“We had our ESTHER Strategy Days. It was once a year that we had a really big gathering about what we are going to focus on. And we invited managers, we invited the coaches, we invited Esthers. So, one-third of the group [of 30] were Esthers and the other were working in health and social care. And, for me, that was a very big success, but it also became a threat. So, they took it away because they said you can’t have strategy day because you are not a manager. So, we changed the name. Now we have the ESTHER Inspiration Day”. [JKG-01]

The implementation process has been incremental and iterative to balance the grassroots pressure for innovation with the internal resistance to patients as equal partners, while ensuring real change results. As an example, in 2007, ESTHER cafes were introduced to connect Esthers and to identify the improvement possibilities most important to Esther. These cafes continue to be held four times per year and have attracted a wide audience, including clinicians and politicians. Esthers share their stories to help leaders and practitioners understand individual experiences, but the process also builds credibility: it requires a check-in with leaders and service providers about what they heard and whether that is consistent with what the storyteller feels is most important, and agreements are reached before the meeting ends about specific action(s) that will be taken to address what is important to Esthers.

“When we listen to a story, we ask the group, ‘What did you hear?’ And we are trying to confirm whether we are hearing different things than [what] Esthers really mean. So, the staff sometimes think, ‘This is very important’. But when we give that back to Esther, she says, ‘Well, that’s not so important for me. For me, this is important’. So, the ESTHER cafe is an activity to identify improvement possibilities. That’s one of the activities”. [JKG-02]

Assimilation: By 2016, ESTHER had evolved from being a network to becoming assimilated as a mindset – the central concept driving innovation in the system in the Jönköping Region. By this time, the decision was made to withdraw funding specific to ESTHER other than to support coach education and to have no single person responsible as leader, as it is intended to be fully assimilated as part of the normal way of working. At the same time, without dedicated funding and leadership, questions remain about sustainability.

“As I said, it is a mindset. Now it is implemented in these programs – the question: ‘What’s best for Esther?’– you will find you can’t find one person who is responsible for ESTHER in Sweden, but there is a programme group and the programme group is trying to find out ways how to spread it in the whole region, because we have some difficulties there. It’s a mindset and it should be part of the daily work. And we are getting there. I think it’s very much dependent who is leading all these kind of leadership programmes, and do they really take the ESTHER philosophy to heart?” [JKG-02]

At this point, all steering groups were removed, being seen as no longer necessary. This removal of infrastructure (formal structures, funding) initially concerned committed leaders, with a risk of co-optation of the ESTHER concept without true adherence in practice. However, there was a widespread sense among interviewees that the ESTHER philosophy has been assimilated as a core value that continues to influence all activities, permeating the culture to become the routine practice in Jönköping.

“It’s a very normal mindset in one of our hospitals to ask the question, ‘What’s best for Esther?’ That’s just a normal way of working and people are just using that word and that question”. [JKG-FL-01]

See Fig. 1 for a summary of the Case 1 adoption, implementation and assimilation timeline.

Case 1 ESTHER coproduction adoption, implementation and assimilation timeline

Historical context: Making Recovery Real gives people with lived experience of mental health difficulties the opportunity to be at the centre of decision-making, service design and practice development in the community of Dundee, Scotland by changing the terms of the dialogue about recovery, mental health and well-being. It began in 2015 as a collaboration of 10 local public, voluntary and community organizations who responded to a call from the Scottish Recovery Network (SRN) to work together to take a new approach to improve the experience and outcomes for people living with mental illness. Initially, the partner organizations endeavoured to develop and deliver more recovery-focused policies and practice by centring lived experience in answering the question: “How can we make recovery real in Dundee?” They brought together interested people, including those with lived experience, at collaborative cafes; a series of events where priorities and accompanying actions were identified, and where participants were equal contributors to the process and its outcomes. To foster the integration of lived experience into system design and practice, the priorities identified were to (i) collect and share recovery stories so that lived experience is at the core of service design, delivery and practice; (ii) develop peer support roles and training; and (iii) celebrate recovery [ 24 ].

Adoption: In the external context , the mental health system remained dominated by the medical model, a lack of system innovation and acute services prioritized over community services. Yet, recent Scottish health and social care system integration has supported partnership working. Simultaneously, SRN, a national voluntary organization established in 2004 to promote recovery principles within the mental health system, was shifting from working with the National Health Service towards building coalitions of change within communities and a whole-systems approach to promoting recovery. SRN solicited proposals from local groups and organizations, offering their support for community-based collaborations that would involve people with lived experience in developing local initiatives to support mental health.

Factors in Dundee’s internal context also converged to support a proposal put forward to SRN for an innovative approach. First, the Dundee Third Sector Interface (TSI), which supports the representation of third sector organizations in local authority planning, had been working to better involve people with lived experience in mental health system planning, and meetings with their network members were becoming more recovery focused. A recent inquiry into mental health services and a fairness commission on poverty (a longstanding local issue) also motivated the local council and Health and Social Care Partnership (HSCP) to take innovative action focused on prevention versus mitigation.

“And I think the Health and Social Care Partnership realized that they needed to do more than mitigation … they have been really, really clear on the need for new ways of doing things for about the last 10, 15 years”. [DND-02]

Furthermore, Dundee City was preparing to develop a new mental health strategic plan and, in the hope of influencing the strategic priorities and the future approach to engagement locally, the TSI brought partners from across community services, the local authority and representative groups who had been attempting to make change in the system to submit a proposal for SRN’s support. Individual leaders from within the partner organizations, motivated by their own lived or professional experience, were drawn by the innovation’s features : to support any concerned citizen to contribute their inherent resources through meaningful involvement and an asset-based approach:

“… So lived experience is essential, bringing people together, involving everybody who wants to be involved in each aspect of the process; so, firstly in agreeing what it is they want to achieve, then in making sure that it is carried out, also in having an actual role in actively carrying it out, so not just identifying things other people should do but having a vested interest and an active contribution to the activities that are going to be – whatever it is that’s going to be done differently, basically”. [DND-04]

SRN acted as a change agency, helping to alleviate tensions among the coalition and supporting their process of exploring the opportunity and submitting a successful proposal.

Implementation: First, SRN helped to bring the individuals involved together to establish a shared vision for the process among the local integration bodies (TSI and HSCP) and a TSI-supported service user network, reducing competition among the service provider partners. Within the inner context of the partnership, there was a commitment to coproduction processes and peer support as a critical opportunity to incorporate more lived experience into the mental health system. Despite these efforts, some of the original partners could not align themselves with the experience-led approach and discontinued their involvement knowing they could return at any time. Undaunted, the remaining partners proceeded by working with the “willing”, beginning with increasing local knowledge of recovery approaches and exploring what recovery meant to local citizens.

“… at the very start, it was a case of, ‘Right. We don’t really know where we want this to go. And actually, are we the ones to be dictating where this should go? No, we’re not. What’s most important is that we’re listening to people with lived experience, people on the ground, and they should be the ones that are telling us what needs to be changing’. So from the beginning, the sort of first step was looking at how we can engage with local people. And we were really keen to make sure that it was meaningful … And we thought this involvement can’t be tokenistic. People need to be on board, and it needs to be collaborative from the start”. [DND-05]

To build connection and trust between participants while shifting to a peer-led approach, the implementation process involved facilitating a series of coproduced, discussion-based events where people with lived experience were invited to be involved in all stages from planning and executing the events, to identifying and achieving priorities. The role of professionals shifted to “being on tap, not on top” [DND-02]. SRN provided developmental support to the Dundee partners to deliver the events, the features of which were welcoming and inclusive, avoiding formal presentations in favour of fun, health-promoting activities that allowed community members to feel heard, and demonstrated alignment with their own ideas and values.

“… what we did—and I would say I think that really set the tone – was rather than have lots of presentations, what we did was, at the event, we welcomed everybody, but we invited lots of the groups to run taster sessions of the things they did. So, that actually brought a lot of people with lived experience because they were coming along to demonstrate their finger painting. There was hula-hooping. There was wellness action planning. There was how to sleep well [sessions]. And in every corner of this venue, there was little groups of people who were painting pebbles, things like that. And then in the afternoon, we had a big conversation happen, world café style. And the sort of comments we got from people were, ‘I felt this was my event. This was for me. It wasn’t for them, the professionals’”. [DND-02]

From these discussions, it emerged that understanding local experiences of personal recovery was the most preferred and effective conveyor of local knowledge and motivator for change for the range of stakeholders. Storytelling became the primary vehicle for relationship building. Peoples’ stories were compiled into a film that premiered at a well-attended, prestigious 'red-carpet' event at a local cinema house, and subsequently became a tool to foster collaborative conversations at engagement events.

“And the film galvanised things and I think because we’d moved beyond that individual telling their story to having a 20 minute film of people reflecting on recovery, which is quite different from telling a story, say, of illness”. [DND-02]

The film drew strategic attention to MRR. This culminated into a consensus to embed recovery, backing for continued peer support and recovery work into the new Dundee Mental Health Strategy and accompanying action plan.

Assimilation: The MRR partner organizations have adopted a peer-led approach to their efforts to promote mental health recovery going forwards. Partners are also now far more involved in collectively determining the distribution of funding through the HSCP and in designing new mental health services.

Locally, the MRR approach has also been included in the Dundee Mental Health Strategy, granting the third sector more influence and collective power in local health and social care planning. The adoption of the MRR approach by the Dundee HSCP has strengthened the importance of mental health locally, dovetailing with the recommendations of the independent inquiry on poverty. At the national level, a Scottish government funding programme to increase the number of mental health workers in community-based services provided an opportunity for the HSCP to fund additional peer support roles, a key initiative within MRR.

Overall, the MRR partnership can be said to have had a transformative effect locally. It has led to better working relationships between providers and continues to drive progress. Furthermore, lived experience is being built into the system infrastructure through actions prioritized in experience-centred collaborative conversations: expansion of the local peer recovery network, development of peer support roles, implementation of peer-led services, peer support training provision and building recovery awareness. A key feature of ongoing progress has been that lived experience partners have been able to move in and out of active participation roles throughout the process, as their recovery journeys and contexts have allowed.

“There was that sense of collaboration that continued ... We kind of all came together to discuss how we felt our organizations could contribute to that bigger picture and the strategic objectives moving forward, and not just the strategic objectives in relation to Making Recovery Real but the wider kind of city and what they were looking for in relation to the local mental health strategy and the city plan”. [DND-05]

Participants describe the process as a difficult yet joyful and rewarding journey. For some organizations, the introduction of the MRR approach has motivated significant recovery-oriented change in their values and structure, further cementing system-level impact.

“Making Recovery Real has really been – I suppose we’ve adopted the principles and approaches … We try to adopt those as far as possible in all of our work. And we don’t badge it all Making Recovery Real, but we use the learning from it, I would say, in everything we do now, everything in the programme”. [DND-04]

See Fig. 2 for a summary of the Case 2 adoption, implementation and assimilation timeline.

Case 2 Making Recovery Real coproduction adoption, implementation and assimilation timeline

Historical context: CMHA Learning Centres began development in Manitoba in 2015 as a coproduced adaptation and renaming of Recovery Colleges, which originated in England in 2009 with a focus on people with lived/living experience of serious mental illness. The aim of Recovery Colleges is to bring the lived experience of people with mental illness and other community members together with professional expertise to locally plan, develop and deliver educational courses about mental health and recovery, with the aim of empowering people to support their mental health and well-being. The concept of recovery education originated in the USA [ 25 , 26 ], and before adopting the Recovery College model, CMHA Winnipeg had offered psychosocial rehabilitation (PSR)-based recovery education since the early 1990s. In 2015, the CMHA Winnipeg branch leader conducted an internal evaluation of this programming, which suggested that improvement was needed to meet the psychosocial health and well-being needs of the community. Around the same time, the new leader of the CMHA Central branch in Portage la Prairie, Manitoba sought a fresh approach to its clubhouse programme, a mutual support drop-in centre, in response to member feedback. Leaders and service users of both branches embraced the Recovery College and coproduction approach to better meet client needs. CMHA Learning Centres build on the Recovery College principles, with the programming and the target audience expanded to promote living well among the broader population, as well as recovery education for people with lived experience of mental illness. The CMHA Central branch’s Thrive Learning Centre and the CMHA Winnipeg and Manitoba branch’s Well-being Learning Centre opened in September 2017 and January 2018, respectively.

Adoption: In the external context , the national policy context was supportive of a recovery and well-being approach; it was the focus of consultations over the 2008–2012 period prior to the release of Canada’s mental health strategy [ 27 ]. This enabled Manitoba bureaucrats to pressure provincial government leaders to cosponsor a 'Recovery Days in Mental Health' conference held in Winnipeg in June 2015. An English Recovery College champion was a keynote speaker and sparked interest in the model among CMHA branches in Manitoba. The Winnipeg Regional Health Authority (RHA), the major funder of the Winnipeg CMHA branch, also supported recovery and mental health promotion approaches. Informants reported that Manitoba’s culture of innovation and solidarity, with its many small rural communities, also aligned with the coproduction philosophy of inclusive innovation.

In the internal context , the Recovery College model resonated with existing branch cultures of deep commitment to recovery-oriented work and strong peer support foundations. CMHA’s federated structure allowed each branch autonomy to develop its own programming, with support from a national office. Attractive innovation features were the existing evidence base, emphasis on lived experience through coproduction in course development and facilitation, opportunity for student skill building, and flexibility to accommodate local needs and strengths. The instructional climate was also appealing, as it could offer people with lived experience a sense of community and could promote their self-efficacy and confidence while reducing the power imbalance and fostering relationships between staff and students. The Recovery College model could also offer a more immediate response in terms of educational support to people needing care and facing long wait times for traditional services.

“I would say there’s probably many other things besides instruction. I think there’s relationship-building that happens so there are connections between students and between the facilitators and the learners. It’s the development of a space that allows for people to develop skills that are unrelated to the content. So, people also learn skills like sharing in a group context, so confidence-building, self-efficacy. When you can cultivate a skill in one area, you build confidence, and you start to believe that you have the ability to learn and to develop new skills. So that sense of self-efficacy is very integral to the recovery and well-being journey”. [WPG-02]

The importance of individual characteristics was demonstrated as passionate leaders in the Winnipeg and Central branches who were committed to advancing upstream mental health promotion and PSR were impressed by the model and together, they researched it further to inform adoption decisions. The coproduction process aligned with CMHA’s “nothing about us without us” approach and could foster a sense of ownership. In both branches, the name Recovery College was changed to Learning Centre during the adoption process, which better resonated with community and agency participants.

Implementation: In the external context , in early 2017, CMHA Winnipeg and Central branches met with CMHA National to implement Learning Centres. Although no new funding was made available by the RHAs, philosophical support enabled the repurposing of existing funding for recovery education and peer support. In 2018, CMHA National and CMHA Winnipeg leadership visited England to meet recovery-focused mental health services experts and to see the model in action. This visit was crucial in fostering strong relationships between the model initiators and CMHA leaders who discovered common visions to widen the target audience to anyone in the community interested in mental health issues, thereby making mental health a universal concern and promoting a living well approach. Collaboration with an Ontario-based psychiatric hospital, with similar values and interest in Recovery Colleges, supported programme evaluation to produce evidence of effectiveness.

Internally , the Winnipeg and Central branches collaborated on initial model and course development, and took a staged approach to opening their Learning Centres. In the Central branch, where resources were tighter and there was a large geographic area to serve, creative approaches to leverage local support and assets were used. Health professional placement students supported the small branch to prepare for launch and in doing so, encouraged staff buy-in. Another peer service organization provided funding support and this, along with community grants, covered staffing, technology, social marketing and other costs that are traditionally not eligible for provincial funding.

“[A] critical moment would be the establishment of a partnership. I think that was a critical moment. I walked away and I know my staff did, too, with an immense sense of relief after I could tell them that [a peer Manitoban mental health community organization] was on board to help make this a reality”. [PLP-22]

The Winnipeg branch also leveraged internal resources, including an existing peer support group whose members assisted in developing the first five courses.

“And so we actually relied on some communities that existed within our CMHA. So we had a group of individuals who are peer supporters to one another. They had taken our workshops in the past. And then they created, on their own, their own support group, and designed that support group based on their needs and on an educational focus. So we actually asked them if they would be our initial coproduction group”. [WPG-04]

The passion of individual CMHA staff and leaders, many with their own lived experience, made them champions who demonstrated their commitment to valuing expertise derived from lived experience. These individuals also helped build the external linkages with organizations and key people both nationally and internationally. Innovation features allowed for initial small-scale implementation, leveraging local assets and community strengths before expanding further. The flexibility to offer “something for everyone” and promote “living well in your community” garnered broad interest and unanimous buy-in from community members. The flexibility of the model also allowed the Winnipeg branch to retain PSR influences from their colleagues at Boston College.

The collaborative coproduction process fostered a sense of ownership, friendship building, balance across perspectives and acceptance within the classroom. This affirming process allowed room for creative input and for trial and error, with the process itself evolving to become more effective over time. It also facilitated the expansion of course offerings, as students were encouraged to lead future course development. Accompanying changes to the physical space and staff roles helped in welcoming the whole community, meeting the needs of vulnerable groups in society and addressing access barriers.

Assimilation: The Central branch has been unable to coproduce new Learning Centre material during the COVID-19 pandemic, yet it continues to offer its existing content. The Winnipeg Learning Centre was able to shift to virtual and then hybrid online and in-person coproduction activities, while ensuring fidelity to the core Recovery College principles.

“And some of the other things that are in the fidelity assessment are: Are you recovery-focused? Are you community-focused? Are you collaborating with the people who are consuming your services? So, it’s a really easy fidelity to conform to but also have room to be kind of creative because they’re not dictating what courses you should have. The fidelity is that you provide courses”. [WPG-04]

In Winnipeg, the Learning Centre continues to expand and evolve, and is reported to have had a gradual but transformative impact on organizational context and values within the branch, by providing a universally accessible platform that demonstrates the value of engaging people with lived experience at every step. The coproduction approach to course development has ensured that content remains current and relevant through creativity, diversity and responsiveness to people’s needs. Leaders’ commitment to the model and ongoing evaluation to ensure it is meeting local needs have supported wider assimilation of coproduction approaches in other branch programming as well. New leadership in the Central branch has expressed the desire to revive the Learning Centre’s coproduction activities.

See Fig. 3 for a summary of the Case 3 adoption, implementation and assimilation timeline.

Case 3 CMHA Learning Centres coproduction adoption, implementation and assimilation timeline

Cross-case comparison

Adoption: Shifting ideas in the public policy realm and supportive external change agents created a conducive external context . In Cases 1 and 2, shifting ideas pertained to interprofessional and intersectoral collaboration and in Case 3, national and provincial discussions about a recovery and well-being orientation were important precursors to coproduction with people with lived experience. Internally , tension for change was evident in all cases; however, the process by which this unfolded differed, as a natural progression of ongoing improvement efforts in Cases 1 and 3 and as a deliberate response to an opportunity created by an external change agent for local system-wide transformative change in Case 2. In all cases, passionate individuals , many with their own lived experience, and a philosophical approach that resonated deeply and widely was a core feature leading to adoption (see Table  6 ).

Implementation: In all three cases, building local partnerships and/or networks in the external context was integral to implementation. These partnerships and networks helped to overcome internal resistance within existing power structures (Case 1), created a community coalition that could move forwards in the face of resistance within traditional mental health services (Case 2), and offered material support and expertise to support implementation (Case 3). In Cases 1 and 2, there was no 'programme' per se, rather a philosophy steered by guiding questions, and in Case 3, the Recovery College model itself was designed to realize its embedded philosophy through coproduced educational programming. These features drove a micro-level movement for change (all cases) that was locally adapted, for example, to become “something for everyone” (in Case 3). Philosophical alignment also helped in building trust across collaborating organizations to support implementation and as a shared foundation for overcoming differences during implementation. Implementation proceeded incrementally at the grassroots level in all cases and by working with the willing (see Table  7 ).

Assimilation: There have been different forms of assimilation across all three cases, with transformative impacts not only on the organizations involved but with impacts extending to the broader organizational and political context. A widely embraced mindset in the region, new structures and a growing international network (Case 1); impact on the local mental health strategy and continuing transformative effects on partnerships among community agencies (Case 2); and assimilation to other programmes and branches (Case 3) are some of the ongoing transformative impacts.

In Case 3, assimilation was characterized by customization, as both branches have changed the name and broadened the reach of Recovery Colleges, while maintaining fidelity to core principles. At the same time, challenges to sustaining such transformative change going forward were a concern without targeted leadership and funding (see Table  8 ).

The analysis of these cases of adoption, implementation and assimilation of innovation demonstrates a range of factors from existing frameworks that shaped the stories of these coproduced innovations. The analysis also suggests additional considerations beyond established frameworks when aiming to engage structurally vulnerable people in coproduction activities that can help to overcome structural barriers and address power differentials in legacy systems.

Existing frameworks and models were very helpful in pointing to the interplay between the many factors operating at different levels in each context. These comprehensive frameworks provided a wide lens that was useful for thoroughly investigating different contextual elements. However, at times, this comprehensiveness made it difficult to tease out the essential causal story from our data to understand how each set of coproduced innovations emerged [ 28 ]. In our analysis, existing frameworks were most helpful when comparing across cases to identify overarching patterns, such as the influence of shifting policy ideas and external change agents in the external context during adoption and the role of community partners and network building in the implementation phase.

At the same time, particularly compelling considerations involving structurally vulnerable groups identified here were less evident in existing frameworks. Notably, there were two important differences in the nature of the ‘programme’ in this context. First, existing frameworks suggest a predefined 'programme' to adopt; however, there was no predefined programme per se in two of our cases. Instead, change was more ideological/philosophical in nature, captured simply by a set of guiding questions (two cases) or embedded as a central feature of an existing program with lots of room for customization (one case). The central philosophy in these cases corresponded to efforts to raise the profile of traditionally marginalized voices by shifting normative paradigms about what types of knowledge (for example, lived experience) and whose voices (for example, structurally vulnerable service users) should be heard in traditional systems. Second, the process (coproduction) could not be disentangled from this essential philosophy and, in some cases, it was met with considerable resistance. Including vulnerable people as genuine partners in coproducing innovations was perceived as a 'threat' to some managers (Case 1) or to the prevailing orthodoxy of 'Quality Improvement' (Case 2).

These 'programme' features suggest a second consideration in terms of implementation processes . The clear intention to shift the existing power balance in systems and within organizations needed a set of resources that went beyond the capacity of any one organization. While high-level leaders with their own lived experience were instrumental in providing vision and support, the implementation process relied heavily on relationship building across partner organizations and networking at the grassroots levels rather than on top-down directives. Meaningful service user involvement was considered critical in making transformative service and system culture change, often disrupting traditional structures, networks and communication. Shared values, the development of a group-based belief system, core activities and a different relational environment and leadership [ 29 , 30 ] are central to social movement theories. Furthermore, the definitive objective of stepping outside organizations within the formal healthcare system to instead derive a new way of working across many community organizations led by people with lived experiences is not clearly captured in existing frameworks, which typically speak to innovation within existing structures of power in organizations and systems.

Finally, the cases analysed here suggest important differences in temporal dynamics at play that were not elaborated in existing models. Consistent with concepts of change in complex adaptive systems and theories of policy path dependence and agenda setting, adoption could occur through a slow internal tension for change that built over time and culminated in coproduction as a natural evolution of ongoing improvement efforts or through seemingly sudden 'transformative' reform where a confluence of interested groups came together in the face of an opportunity to do something differently. Ideas about change in complex adaptive systems such as emergence, self-organization, adaptation, change over time, distributed control and tipping points [ 31 ], and from policy literature such as path dependence [ 32 ], multiple streams theory [ 33 ] and distributed control could be informative in this respect [ 34 ]. Our participants suggested that because each case relates to a set of concepts and principles that were collectively generated over time, there was a need to better understand this process as it unfolded.

While existing models were helpful in considering a wide range of factors to consider and recent updates suggest a movement away from concepts such as 'programme' to 'innovation' [ 19 ], the temporal, relational and power dimensions discussed here were validated by our collaborators as equally important considerations. Exploring these dimensions will be the focus of future work.

Limitations and future work

This work is subject to several limitations. First, it is based on a case study of three examples of coproduction of health and social care innovations in different national contexts in the northern hemisphere. The findings may not be transferable elsewhere. Furthermore, when considering our findings in relation to the CFIR, DOI and assimilation frameworks, it is important to note that these frameworks were not specifically developed for an innovation process involving service users at all stages of innovation adoption, implementation and assimilation. However, the limitations in adopting and applying these frameworks here have led to a careful examination of what is unique to coproduction processes involving vulnerable populations. A forthcoming contribution will try to capture these unique elements and position them within the innovation, power, and social movement literatures. Finally, the analysis here is primarily based on our 'wave 1' home site findings from this longitudinal case study, and new insights may be gained from a deeper evaluation of our wave 2 and wave 3 findings. The latter pertain to processes of ongoing coproduction in practice and diffusion to other contexts, respectively, and will be analysed in forthcoming work.

While our case study was extremely helpful in identifying core considerations for factors influencing the adoption, implementation and assimilation of three cases of coproduced health and social care innovations, several nuanced considerations when applying existing theoretical frameworks in the coproduction context emerged: the nature of the 'intervention' being a philosophy rather than a concrete set of steps, the intertwining of intervention and process and the need to study evolution of the intervention itself as it emerges over time, greater attention to partnered processes as disruptors to existing power structures and an emphasis on driving transformational change in organizational cultures. Future work will explore these considerations further.

Availability of data and materials

The datasets generated and/or analysed during the current study are not publicly available due to the study’s small sample size and the key informants’ roles as leaders within small organizations, making it difficult to deidentify their data. However, the datasets are available from the corresponding author upon reasonable request.

In some cases, these individuals also had lived experience of vulnerability that also motivated their work, but this was not a specific requirement for study participation.

Abbreviations

Consolidated Framework for Implementation Research

(SingHealth) Centralized Institutional Review Board

Canadian Mental Health Association

Diffusion of innovation

Health and Social Care Partnership

McMaster University Research Ethics Board

Making Recovery Real

Portage la Prairie

Psychosocial rehabilitation

Regional health authority

Scottish Recovery Network

Third sector interface

United States

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Acknowledgements

We wish to thank all participants in this study for giving their time and for sharing their experiences. We also thank the study’s collaborators who provided important background to the cases contributing to the research design/direction, acted as local gatekeepers to the cases and/or who helped to interpret the data. Over the life of the research project, the collaborators have been: Louise Christie (Scottish Recovery Network), Marion Cooper (CMHA Manitoba & Winnipeg), Olivia Hanley (formerly of the Scottish Community Development Centre), Greg Kyllo (formerly of CMHA National), Erica McDiarmid (formerly of CMHA National), Susan Paxton (Scottish Community Development Centre), Denise Silverstone (CMHA National), Stephanie Skakun (CMHA Manitoba & Winnipeg) and Nicoline Vackerberg (Region Jönköping County). Without their involvement, this study would not have been possible. Finally, we thank Sophie Sarre for her contributions to wave 1 interviewing and early coding framework development and data coding.

This manuscript draws on research supported by the Social Sciences and Humanities Research Council Partnership Development grant no. 890-2018-0116. The funders had no role in the design of the study; in the collection, analysis and interpretation of data; or in writing the manuscript.

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GM, GR, ML, SK and SM conceived of and designed the study. GM, JG, NV, EL and SC collected and analysed the data under the guidance of GM, GR, ML and SK. GM, JG and PH interpreted the data. GM, GR, JG and PH drafted the manuscript. ML, NV, SK, SM, EL and SC reviewed and commented on different versions of the paper. GR, GM and JG revised the manuscript following peer review, in consultation with the other authors. All the authors have read and approved the final manuscript.

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Research ethics clearance was obtained from the relevant academic research ethics boards (McMaster University Research Ethics Board [MREB Project ID 2066], Aston University Ethics Committee [Rec Ref #1611]; King’s College London Research Ethics Office [Reference Number MOD-19/20-17350]; SingHealth Centralised Institutional Review Board [CIRB Ref# 2020/2341]; and Swedish Ethical Review Authority [Etikprövningsmyndigheten, Dnr 2019-06373]), and in light of this, ethics review was waived by the boards of the collaborating organizations (Canadian Mental Health Association, Manitoba & Winnipeg branch, the East of Scotland Research Ethics Service). Participants received letters of information outlining the study objectives, protocol and risks prior to consenting in writing.

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Additional file 1..

Provides additional details about the sampling frame (that is, the organizations the interviewees are associated with, the document titles and types).

Additional file 2.

Demonstrates how concepts from the CFIR, DOI, and compatibility gaps frameworks were incorporated into the coding framework.

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Mulvale, G., Green, J., Robert, G. et al. Adopting, implementing and assimilating coproduced health and social care innovations involving structurally vulnerable populations: findings from a longitudinal, multiple case study design in Canada, Scotland and Sweden. Health Res Policy Sys 22 , 42 (2024). https://doi.org/10.1186/s12961-024-01130-w

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Barriers and facilitators of the reporting by family doctors of cases of domestic violence – a qualitative study across Portugal

• Diana Nadine Moreira 1 &
• Mariana Pinto da Costa 1 , 2 , 3  

BMC Primary Care volume  25 , Article number:  107 ( 2024 ) Cite this article

Metrics details

Domestic violence (DV) is a serious and prevalent public health problem with devastating consequences for the victims and their families. Whilst the number of cases reported to the authorities has risen in recent years, many victims still chose not to present a complaint. In Portugal, to address this, DV became a public crime. As victims of DV present multiple health problems and frequently seek professional help, family doctors are in a privileged position to detect and report cases of DV to the authorities. However, little is known about what motivates these professionals to report or not the DV cases they encounter in their practice to the authorities.

We conducted semi-structured interviews with family doctors from all regional health administrations of continental Portugal. Interviews occurred between July 2020 and September 2022, were conducted in person or remotely, audio recorded, transcribed, and analysed using thematic analysis. Content analysis was conducted to assess the agreement or disagreement regarding mandatory reporting in each of the themes and subthemes.

Fifty-four family doctors took part in this study ( n  = 39 women, n  = 15 men). The main themes that arose from the analysis were: “Barriers related to the physician’s activity,” “Barriers related to the victim or aggressor,” “Facilitators related to the physician’s activity,” “Facilitators related to the victim or aggressor.” Although different barriers were described, most doctors agreed with the mandatory reporting of DV cases.

Conclusions

Family doctors encounter multiple barriers and facilitators when considering reporting a DV case to the authorities. The results of this study can help develop new interventions to address the barriers described by the doctors, increasing their compliance with mandatory reporting, the protection of victims and the just persecution of the aggressor.

Peer Review reports

Domestic violence (DV) is a serious social and public health problem, highly prevalent and globally widespread [ 1 ]. It encompasses any act or threat of physical, psychological, or sexual violence perpetrated within the context of an intimate relationship [ 2 ]. The World Health Organization estimates that one in every three women experiences physical and/or sexual violence during their lifetime [ 3 ]. Among male victims’ the prevalence varies between one in every four to one in every ten men, according to different studies [ 4 , 5 ].

In Portugal, DV is the most frequently committed crime [ 6 ]. During the first semester of 2022, the Commission for Citizenship and Gender Equality based on the numbers provided by the Public Security Police (PSP) and the Republican National Guard (GNR) registered 14363 DV reports to those law enforcement entities, which correspond to 2192 more cases compared with the same period in 2021 [ 7 ]. It is thought that most cases of DV are never reported. The Portuguese Support Victims Association (APAV) assists victims of multiple crimes. From the 25838 support requests presented to APAV in 2021, 76.8% ( n  = 19846) were related to DV. However, only 46% ( n  = 6067) of the total of request were reported to the police [ 8 ].

To address the number of reports, provide victims protection, and ensure the persecution of the aggressors, DV became a public crime in Portugal in 2000. This means that anyone can file a DV report, which is enough to progress the criminal proceedings, even against the victim’s will. The facultative character of the report applies to the general population but in the case of public workers, such as policemen and health care professionals it is a professional obligation. These mandatory reporting legislations have been regarded as controversial policies, with limited studies to support their efficiency and applicability [ 9 ].

Family doctors provide continuous follow-up of their patients and families, sometimes through several years, developing a close relationship with them. The characteristics of family medicine place family doctors in a particularly privileged position to identify, refer, and report DV cases they may encounter in their clinical practice. DV victims’ resort to health care services more frequently than the general population presenting multiple health conditions as a result of acute or chronic abuse [ 10 , 11 , 12 ]. These contacts with the health system provide important opportunities for intervention, and according to mandatory reporting legislations, these should result in a report to the authorities. However, little is known regarding the degree of compliance with the law by family doctors and what motivates them to present, or not, a report to the authorities.

Therefore this study aimed: i) to assess the barriers and facilitators that make family doctors in Portugal to present, or not, a report to the authorities of a DV case they encounter in their clinical practice, and ii) to assess the agreement or disagreement of family doctors in Portugal with mandatory reporting of DV in association with the factors influencing their decision to present or not a report.

Study design

We conducted a qualitative study based on semi-structured interviews with family doctors in Portugal. The interview guide was developed by the authors. Prior to the interview, participants’ sociodemographic data was collected using an online questionnaire.

Setting and participants recruitment

This study was conducted in the five Regional Health Administrations (RHA) of continental Portugal: North, Center, Lisbon and Tagus Valley, Alentejo and Algarve. An email was sent to every Family Health Unit and Personalized Health Care Unit of each of the RHAs. This email introduced the study, inviting family doctors of each institution to take part. It also contained the email address of the lead author (DNM) and a link that would direct the potential participants to an online page containing the information sheet with additional detailed information about the project, the informed consent and the sociodemographic questionnaire. This page was also promoted in several social media groups and online forums for family medicine doctors. At the end of the online questionnaire, it was requested the participant’s email to enable further contact and the scheduling of the interview. The interviews were conducted by the lead author (DNM) through online platforms: Zoom, Skype, or Teams, or in person when possible. The only inclusion criteria was to be a specialist in family medicine currently working in continental Portugal.

Data collection and analysis

The interviews took place between July 2020 and September 2022, and were conducted according to an interview guide developed by the authors. Interviews were audio recorded, transcribed, and analysed through thematic analysis using the approach proposed by Braun & Clarke [ 13 ]. Transcripts were uploaded into QSR International Nvivo version 12, which was used to manage and analyse the data. The analysis was inductive and based on the content of the transcripts rather than on any existing theory or hypothesis. Themes and subthemes were reviewed independently by both authors several times during the analysis process to guarantee internal consistency between their conceptual nucleus and the codes that generated them. Coding saturation was achieved and no new codes emerged from the data. The heterogeneity of the population studied and the broad research question justify the need for a large number of interviews to reach coding saturation. Content analysis was used to assess which theme and subthemes were linked to a favourable or unfavourable position of participants regarding mandatory reporting of domestic violence.

Eighty-four doctors responded to the online questionnaire, after which they were contacted via email by the lead author (DNM). Of these, two changed their mind and declined to be interviewed, two scheduled the interview but did not show up, and twenty-six never replied to the email to schedule the interview. In total 54 participants were interviewed (12 from the North RHA, 12 from the Center RHA, 12 from the Lisbon and Tagus Valley RHA, 6 from the Alentejo RHA, and 12 from the Algarve RHA). Six participants knew the lead author previously to the interview. The median duration for an interview was 23 min (IQR 9:00 to 45:00 min).

The majority of participants were female (72%) and their age ranged from 30 to 65 years. The detailed socio-demographic information of the participants is presented in Table  1 .

The data was organized into four broad themes, each with several sub-themes: 1) “Barriers related to the physician’s activity”; 2) “Barriers related to the victim or aggressor”; 3) “Facilitators related to the physician’s activity”; 4) “Facilitators related to the victim or aggressor” (Table  2 ). These barriers and facilitators are frequently interconnected representing the complexity of DV cases (Fig.  1 : Relations between the themes and subthemes).

Relations between the themes and subthemes

Barriers related to the physician’s activity

Multiple barriers were described with relation to the reporting of domestic violence cases to the authorities that were linked to the doctors’ clinical activity, their fears, and perceptions. One of the first barriers described was the difficulty of detection. In the absence of physical evidence, and when the patient purposely tries to hide, or disguise the causes of their afflictions, doctors’ ability to detect domestic violence cases can be compromised. The interviewed doctors made reference to the multiple signs and symptoms, such as headaches, abdominal pain, and nocturnal enuresis in children that can be related to DV. When the doctor fails to make the connection between these health problems and a possible case of abuse, they may lead to multiple consultations, exams and treatments that fail to address the underlying issue.

“I can’t always understand, sometimes I may suspect something, but if the patient doesn’t tell me anything, does not confirm my suspicion, I’m not always able, and don’t have the time, to pay attention to all the signs, and remember “Wait, this could be domestic violence.”” (Participant 26, Woman)

The difficulty of detection was frequently associated with other barriers such as lack of time and bureaucracies. Several doctors complained about the lack of time in their clinical practice to address DV concerns. They spoke about having a limited time for each consultation, the pressure felt in the waiting room when there is a delay, the high number of patients under their care, and the fact that in most consultations they are expected to address multiple issues and health problems. Doctors also said they did not have the time to deal with the possible increase in bureaucratic paperwork associated with a complaint to the authorities.

“I also want to say that in family medicine we have it all. What we don’t have is the necessary time to deal with all of this. Because there is an excess of bureaucratic work.” (Participant 10, Man)

However, they also express not knowing in reality which bureaucracies and procedures would be implicated. Lack of knowledge was the main barrier to the reporting of DV cases described by the doctors. Some doctors admitted to being uninformed, lacking the knowledge and capabilities to manage, refer, and report a DV case.

“Honestly, I also don’t know exactly which are the procedures. (…) I have a victim here in front of me, who should I call? Honestly, it is a flaw on my part. I have to look into it later.” (Participant 19, Man)

Other doctors, having recognized this gap in their knowledge, tried to look for information regarding DV, but were incorrectly or insufficiently enlightened.

“You can’t. You can’t [referring to the reporting of DV cases]. I asked the juridic department. You can´t. I had that problem and you can’t. Because it is the story that that woman is telling you. She could have, she could have had an injury, she could… you don’t know. And since you don’t know, you didn’t see [the aggression], you can’t.” (Participant 11, Woman)

One doctor described a case of a patient that complained multiple times to be a victim of DV perpetrated by her husband, presenting physical lesions compatible with the abuse described. This husband was known to be an aggressive individual, having assaulted a nurse and the security guard of the health centre. This doctor described that having sought advice from the juridical department of her workplace regarding the possibility of breaking professional confidentiality and reporting the abuse, she was misinformed that she could not report the case unless she was an eyewitness to the violence.

Other doctors described they were misinformed by their hierarchy superiors or by the police department when trying to present a report. This led doctors to request for training opportunities for health professionals and law enforcement regarding DV. Doctors also felt that there is a lack of guidelines regarding DV and reaffirmed the need for the creation and implementation of guidelines and procedures specific to each community.

“There should be an algorithm, something concrete that would tell you, in this case, you must do this, refer to these and that institution, you can present a report using these and that means.” (Participant 51, Woman)

Doctors frequently brought up the fact that they are in a particularly unique, but sometimes difficult position as doctors of the potential victim and aggressor. As family doctors, they are sometimes confronted with two different accounts of the same events, which in the absence of more concrete physical evidence may compromise their readiness to report.

“Because it is like this, you rarely see any physical marks. Even a belt or something like that… it’s what people tell you (…), we don’t know what is true, we hear people talking, but we don’t know what is true, and then you have the other side of the story. Since we are a family doctor we have both components, and then sometimes as well… I’m not a cop! To clarify the situation. And I don’t always believe it.” (Participant 39, Woman)

This fear of false testimony is frequently associated with other barriers such as the fear of breaking the doctor-patient relationship, either with the victim or the aggressor, especially if unjustly accused. Doctors impart great importance on their ability to establish a therapeutic relationship with their patients and fear that a report could break their trust compromising future interactions.

“It is complicated to keep the relationship. Because you can never again establish a level of trust if you make a report. Who is gonna trust us? Neither the aggressor nor the victim. Especially the relationship with the aggressor is destroyed”. (Participant 22, Woman)

A doctors-patient relationship is based on multiple assertions, one of which is the duty to keep the doctor’s patient confidentiality and professional secrecy. This secrecy is a right of the patient recognized by law and can be perceived as a conflicting concept with mandatory reporting.

“Another question is doctor’s patient confidentiality, isn’t it? It all depends if the victim wants to present a report, and that isn’t a problem, right? Now if they inform me in confidence that they don’t want anyone to know, I have an obligation, by law, as a healthcare professional, to keep it secret. Then we enter a very sensitive area.” (Participant 35, Woman)

Doctors also expressed concerns regarding the possible legal consequences of breaking doctor-patient confidentiality. This idea is included in another subtheme, fear of retaliation against the doctor. Doctors feared being reprimanded by their hierarchy superiors after a report, being sued for defamation, becoming a victim of physical violence by the aggressor, and seeing their reputation slander as an act of revenge. Many of the doctors interviewed cited that there is no security in their health unit feeling completely exposed and unsafe.

“I explained everything [a suspected case of sexual violence of a father against his daughter stated in consultation by the mother] to the investigator, to the inspector, and he told me: “You know doctor, everyone says it’s not true, and right now you are their accuser. They can take you to court for defamation.” I was so choked I could die, as you can imagine .” (Participant 06, Woman)

Doctors also considered that in some cases a report could be non-beneficial, giving priority to other kinds of interventions tailored to the victim and their situation. Doctors expressed that it could be more advantageous to the victim to be referred to support institutions like APAV or Adult Violence Preventions Teams (EPVA) so that the process of reporting the violence to the authorities could be worked out between them and the victims according to their wishes and understanding of their situation. Other suggested means of support mentioned were psychology and psychiatry consultations, referral to a social worker, and family therapy. These alternatives were perceived as more beneficial, especially in cases where the aggressor was violent as a result of a mental health disorder.

“I think that in the case of this family reporting the case will not help, it will not be helpful for the police to go there, it’s not gonna help the family. Maybe as the family doctor, with the help of the psychologist, something more based on the family, maybe she [the victim] would gain conscience, and he [the aggressor] will realize that he is abusing her and that it is a crime, maybe if I say that and explained that his wife symptoms are related to the abuse, maybe that could help.” (Participant 45, Woman)

Not only were alternatives sometimes perceived as more advantageous, but also, frequently, the results of reporting the case to the authorities were seen as subpar. The response provided after a report was considered inefficient in protecting the victims from possible retaliation from the aggressor and the punitive measures implemented to correct the aggressor’s behaviour were judged inefficient.

“Is the woman really protected? He is punished in an exemplary fashion? How? In what way? Does he learn, does he not? (…) People told me: “Why would I report? The first time I went to the police and they told me: “Lady! For the love of God. We have better things to do! But then why? What is gonna happen? He isn’t going to jail! It’s no use to complain.”” (Participant 37, Man)

This discontent with the response offered after reporting mostly alluded to the police and juridical system. However, doctors also considered that sometimes the support provided by institutions such as APAV and EPVA was less than ideal.

“I am part of the prevention team. I’m on the Adult Violence Prevention Team [EPVA]. So, I should be more, but it is like… it doesn’t work very well, because we don’t have any time to dedicate to the team. Everything is very, it’s only me and a social worker, and it’s all made in a hurry.” (Participant 17, Woman)

A few doctors also considered that presenting a report was not their responsibility. This position was essentially assumed by the doctors that disagree with mandatory reporting.

“(…) I think that we as doctors should not allow being pushed yet another responsibility, that it is not only ours, as I said it is mine as a person, but I don’t think it is mine as a doctor.” (Participant 08, Woman)

Physical violence was perceived by some doctors as an acceptable response to a cycle of continuous psychological violence, leading the patient to reach a limit, responding aggressively. In these cases where violence was an acceptable response to violence doctors manifested the belief that reporting would not be the answer, preferring to work with the family to cease all kinds of violence.

“Saying that a man hit that woman, yes, he did. But why did he hit her? Could it be that she, for example, was not systematically saying bad things about everything he does ?” (Participant 10, Man)

Barriers related to the victim or aggressor

Different barriers described were related to the victim, the aggressor, and the dynamics of a violent relationship. Doctors manifested a desire to respect the victim’s autonomy supporting her in the decision to make a report. In situations where the victim assumes a proactive stance, working on an escape plan and reflecting on the best moment to abandon the relationship, the doctor takes an expectant position. In these cases, premature reporting done by the doctor could be detrimental putting at risk the victim’s plan.

“When the person shows that she has a plan, that she is in control of the situation and is taking measures to resolve it. In that case I will support her but I will not interfere.” (Participant 34, Man)

Doctors’ perception of the degree of violence is a concept that can be seen either as a barrier or a facilitator of reporting. It encompasses the doctor’s evaluation of the gravity of violence suffered by the victim, being that in cases where the violence is considered less serious, the doctor will choose not to report it. This perceived “inferior degree” of violence is frequently linked to forms of psychological abuse.

“I knew there was, I know there is verbal violence… I suspected there was physical violence although I never had any evidence. In that case, I didn’t report, but I think that we end up not reporting something that isn’t evidently physical.” (Participant 09, Woman)

Doctors’ perception of the risk incurred by the victim after reporting also impacted their decision in taking action. Doctors reflected on the risk of retaliation and escalation of violence against the victim after a report. This subtheme relates to the idea that the legal response to reporting is not sufficiently protective. During the interviews, doctors recollect several cases where after a report made by the victim, the doctors, or others, the aggressors became more violent.

“(…) If there is a report it can even aggravate the violence or in more extreme cases originate situations that put at risk the life of the victim .” (Participant 46, Man)

The fear of retaliation, among other reasons, can justify the lack of collaboration of the victim, before and after a report has been presented to the authorities. Doctors considered that the involvement of the victim and their willingness to abandon the relationship is essential to the success of the legal procedures and in their absence, doctors felt discouraged to report.

“The victim would go to the emergency department multiple times, but afterwards, since in this case, it was her son [the aggressor], she would end up withdrawing the complaint. So it was very complicated, there were many years of… of difficulties on that point, and talking to the social worker, and following up the case just so she would quit.” (Participant 12, Woman)

Facilitators related to the physicians’ activity

Doctors also reflected on multiple facilitators and motivators that lead them to report cases of DV to the authorities. The possibility of presenting an anonymous report was seen as a facilitator for some doctors by contradicting some of the barriers previously presented. However, some doctors recognize that this method of reporting may be less efficient than when the report is assumed by the doctor.

“The report can be made anonymously. That facilitates. But I don’t know if the report would be taken more seriously if they knew it was a doctor that denounced it.” (Participant 29, Woman)

It is also important to consider that the anonymity of the report does not mean that the victim would not be informed by the doctors of their decision to report. Some doctors consider it important to notify the victim of their intention to report, so she can take measures to protect herself, while others prefer to safeguard their medical relationship with the victim and would not admit to having been the ones presenting the complaint.

Doctors that agree with mandatory reporting laws stated that the knowledge of the case is the only incentive needed to present a report.

“I would tell her [the victim] that it is a public crime, and so, I will have to report it. It is my obligation as a citizen and as a doctor to report the situation.” (Participant 44, Woman)

Some doctors admitted to feeling guilty for not reporting previous cases to the authorities assuming a more proactive stance in following cases. These sentiments arise when the doctors reflect on the risk incurred by the victim in staying in an abusive relationship.

“I remember clearly that she was a little puzzled with that and, at the time, because of my inexperience, I thought I should have taken some measure concerning that family and that child. And even today I regret that I didn’t do anything.” (Participant 13, Woman)

Contrary to the most common perception that the response offered after reporting is inefficient, some doctors shared previous positive experiences regarding the resolution of DV cases, which leads them to believe in the efficiency of reporting.

“I had one patient who was tetraplegic and would tell me that his wife would bite him in the head because it was the only place he could feel it. And, at the time, I called the cops and went in person to present a report to the PSP [police]. That gave me a lot of trouble, they found out it was me, but it had a consequence, the patient started getting domiciliary support. There was surveillance and services were mobilized.” (Participant 39, Woman)

Doctors admitted to having no doubts regarding presenting a report if they were an eyewitness of the occurrence. Even though this is a strong motivator to report, doctors recognize that they are rarely present when violence takes place, and these events do not accurately reflect the majority of cases of DV encountered in their clinical practice.

“That’s what I was telling you, if I was a witness that I was in the front line and saw the aggression taking place, in that case, there is no doubt.” (Participant 36, Woman)

Facilitators related to the victim or aggressor

Doctors also described facilitators of reports related to the characteristics of the victim, the aggressor, or the context of their relationship. In the case of victims who, because of their age, or physical or mental capacity, are unable to protect themselves, doctors agree that the report would be more readily done. Some doctors also cited that the involvement of a fragile individual would motivate them to present a report even in cases where they were not the “direct” victim of the aggression but a spectator. This was especially true in cases where children lived in a violent environment, observing and normalizing the violence between their parents.

“If there are children or elderly involved, people that are dependent, bedridden, with physical… or mental deficiencies, that wouldn’t allow them to protect themselves. In those situations, I think we don’t have so many doubts in reporting it.” (Participant 53, Woman)

Doctors would also be more motivated to report in cases where the victims lack a social or familial network of support, structures perceived by the doctors interviewed as protective to the victims.

“In cases where the person is isolated, when she doesn’t have more family or anybody who she could ask for help. In those cases you have to help [with the report].” (Participant 31, Man)

If the victim requests the doctor to present a report most of the barriers discussed previously cease to exist, being this a motivator for the doctor to take action.

“The victim tells me… that she needs help, she doesn’t dare to do it and she needs help. If so, I would report it .” (Participant 47, Man)

The careful evaluation of the degree of violence and life-threatening risk is considered essential in the assessment of DV cases. Most doctors agree that in extremely violent cases with severe repercussions to the health and well-being of the victim, as well as her eminent security, they would not hesitate in reporting. However, it is important to address that when questioned most doctors confessed feeling insecure regarding their ability to evaluate DV cases, needing more training.

“Situation where I feel, but this is so hard sometimes, that the person is really having her life at risk. Right? Or at risk of suffering damage, substantial damage, I think in those cases there would be no doubts. But sometimes it is so difficult to evaluate to which point… the limit.” (Participant 14, Woman)

Content analysis

All the doctors interviewed were in agreement with the legislation relating to the facultative reporting of DV cases by the general population. However, regarding the mandatory reporting of DV contemplated by the Portuguese law, and applicable to public workers, including those working for the Portuguese National Health System, doctors were almost divided in their opinion with 29 doctors agreeing with mandatory reporting and 25 disagreeing. Using content analysis, we determined which themes and subthemes were more frequently associated with the agreement, or disagreement, regarding mandatory reporting of DV cases (Table  3 ). Overall doctors that agree with mandatory reporting cite less frequently barriers to reporting (147 citations) than doctors that disagree (160 citations). In contrast, facilitators of reporting are cited more frequently by doctors that agree with mandatory reporting (63 citations) than doctors that disagree (48 citations).

Reflecting on the barriers encountered by the doctors when pondering a report, we found an interposition between the doctors that agree and disagree with mandatory reporting, regarding the subthemes: “Lack of knowledge” and “Response perceived as inefficient”. This concurrence between barriers, independently of the position regarding the law, expressed by the doctors shows how these are the greatest challenges faced by doctors when dealing with DV cases being almost universally experienced. Doctors that disagree with mandatory reporting, also frequently cited as a barrier the “Lack of victims collaboration”. These doctors put a great emphasis on the disposition of the victim to corroborate their deposition and collaborate with the legal procedures to achieve the desired response after reporting, considering that in the absence of victims’ approval reporting would be ineffective.

The most common facilitator of reporting cited by the doctors was: “Degree of violence and life-threatening risk”, appearing in 19 of the interviews with doctors that agree with mandatory reporting and 17 of the doctors that disagree. Doctors’ perception of an extreme degree of violence that would compromise their patient security is the greatest motivator for reporting, even between doctors that do not agree with mandatory reporting, limiting their intervention to these and other specific cases, such as those involving a fragile individual ( n  = 11 between doctors that agree with mandatory reporting and n  = 9 between those that disagree). Noticeably, only the doctors that agree with mandatory reporting affirm that the simple “Knowledge of the case” is a sufficient motivator for reporting ( n  = 12 VS n  = 0).

Key findings

This study reports the perceptions of family doctors in Portugal regarding the barriers and facilitators for them to present, or not, a report to the authorities of cases of DV violence encountered in their clinical practice. The four themes that emerged from the analysis of the interviews represent the dichotomic position faced by these doctors and exemplify multiple doubts and concerns felt when dealing with DV cases. The current legislation that recognizes DV as a public crime, making reporting mandatory for public workers, in this case, doctors, is seen as a controversial policy. Doctors agree with the extension of the duty to report to the general population but are divided in their opinion regarding mandatory reporting for health professionals. This division arises from the multiple barriers faced when trying to submit a complaint, especially with respect to the protection offered to the victim and the efficiency of law enforcement and juridical responses. Most doctors also admitted to a lack of knowledge necessary to confidently and correctly manage DV cases, expressing many doubts when approaching a potential victim and not knowing how to follow, refer or report these situations. This lack of knowledge is cited as the result of a lack of training and institutional guidelines. It could also be the result of misinformation spread by hierarchy superiors, law enforcement, and even juridical departments. Independently of their position regarding mandatory reporting, most doctors agree that the involvement of a fragile person in the abusive relationship, and the perception of an extreme degree of violence, compromising the victim’s health and wellbeing, would be strong motivators for reporting the case.

Strengths and limitations

This is the first study that investigates doctors’ agreement with mandatory reporting of DV in association with the barriers and facilitators they encounter when considering to present a report to the authorities. The scarcity of studies on the subject justifies the importance of our findings, which will hopefully serve as a basis for ulterior works and an incentive to debate current practices and policies.

The extension of this qualitative study to a national level allowed the participation of doctors from each of the five RHA’s of continental Portugal, including different perspectives based on multiple social, regional and cultural contexts. This widespread approach enriched our work and is one of the main strengths of this study. However, given that participation in this project was entirely voluntary, it is possible that the findings may not fully represent the views of clinicians with limited interest in discussing topics related to DV.

The use of one-to-one semi-structured interviews was also beneficial since it allowed to investigate with more depth these individual experiences, allowing to emerge sometimes even unexpected themes and subthemes, translating different participants’ experiences.

However, the study presents some limitations. Whether participants had personal lived experience of DV themselves or in their families was not investigated, limiting our understanding of the potential influence of their personal experiences in their opinions and motivations. Furthermore, since the lead author is also a family doctor, this may have influenced the responses from the interviewed participants. The interviewed doctors associated their clinical practice with one of the 5 ARS’s of continental Portugal in the sociodemographic questionnaire, providing data on the geographical location of their workplace. However, it is unclear whether they are working within these entities in the public service, in the private healthcare sector, or in both sectors which is a limitation.

Comparison with the literature

Our study revealed that family doctors in Portugal are divided on their agreement with mandatory reporting of DV. The majority of doctors agrees with the report, especially in circumstances where the victims are incapable of protecting themselves or their health and life is at risk. However, they expressed doubts and concerns that influence their readiness to present a report to the authorities. This conflict of opinions was also observed in a survey, conducted in the USA with doctors working in the primary and secondary healthcare system in California, a state with mandatory reporting laws [ 14 ]. In that study, 86% of the doctors surveyed believed that mandatory reporting increases doctors’ ability to detect and respond to DV cases. However, in this same sample, 56% of the doctors revealed not to comply with mandatory reporting in cases where the victim was against the report. The reasons for this noncompliance with the law were: concerns regarding a possible negative impact of the law on victims’ readiness to look for medical attention, the risk of retaliation by the aggressor, breaking doctors-patient confidentiality, and compromising victims’ autonomy.

The dilemma between reporting, or not, a case to the authorities and the barriers described by the doctors in our study were also present in the literature. A cross-sectional survey conducted in Turkey with primary care professionals showed that the majority of doctors (64.3%) report DV cases encountered in their clinical practice [ 15 ]. When the initiative to report was not taken by the doctor, victims were incentivized to do so. When questioned, doctors affirmed that the reasons to choose not to make a report were: the belief that the victim would choose to remain in the abusive relationship, lack of knowledge regarding detection, documentation, and referral of DV cases, and concerns about doctors’ security. These answers mimic our observations.

The first barrier encountered by the doctors in our study was the “Difficulty of detection”. A qualitative study with telephonic interviews conducted in California, revealed that only 42% of victims share the abuse to their doctors [ 16 ]. However, the stronger facilitator to the disclosure was the direct questioning by the clinician. By questioning the victim in a direct, non-judgemental way doctors can at least in part overcome this barrier. Nevertheless, studies show that doctors rarely question their patients regarding DV [ 2 ]. Particularly, in primary care, less than 15% of patients revealed to have ever been asked about DV [ 16 ]. A systematic review examining doctors’ perceived barriers to screening for DV divided them into five categories: personal barriers, resource barriers, perceptions and attitudes, fears, and patient-related barriers [ 17 ]. Personal barriers encompassed personal discomfort, personal security concerns, and fear regarding misdiagnosis; Resource barriers included lack of knowledge, time constraints, and lack of an office protocol for addressing DV; Fears, perceptions and attitudes were related to the idea that detection DV was not the doctor responsibility, fear of inciting aggressor retaliation against the victim, and concerns about losing their patient trust; and Patient-related barriers, referred to the idea that victims do not want to address the violence choosing to remain on an abusive relationship. It is interesting to note that the same barriers described in this review and faced by the doctors when addressing the victim are also present in our study, influencing doctors’ posture from the initial inquiry to the decision of presenting a report.

Our study revealed that one of the major barriers faced by the doctors in Portugal was “Lack of knowledge”. A Dutch cross-sectional study conducted with 278 mental health professionals questioned them regarding their perception of their knowledge of DV [ 18 ]. It demonstrated that most doctors considered that they lack the necessary knowledge to recognize and support victims of DV. Noticeably, a higher perception of their degree of knowledge did not necessarily correlate to a higher degree of factual knowledge. This observation is similar with our results since some of the interviewed doctors confidently responded to questions based on factually wrong information.

Another major barrier to reporting encountered in our study and expressed by the doctors in Portugal was the perception that the response offered after reporting was ineffective in the protection of the victims and persecution of the aggressors. This viewpoint was shared by doctors in Australia interviewed for a qualitative study regarding child abuse [ 19 ]. In that study doctors described barriers similar to the ones found in our study: lack of knowledge, lack of guidelines and information about reporting, fear of breaking doctor-patient confidentiality, and fear of an incorrect diagnosis.

One of the principal facilitators of reporting described by our participants was the “Involvement of a fragile individual” in the abuse. The majority of studies regarding mandatory reporting focus on more particular forms of violence like children or elderly abuse. However, the barriers encountered seem to be the same. A study conducted in New York regarding elderly abuse, in the context of emergency services interactions, revealed that doctors frequently chose not to present a report due to a lack of protocols and training, difficulties contacting social services, lack of time, and lack of feedback after reporting [ 20 ].

We also found that doctors in Portugal consider the evaluation of the degree of violence as an integral part of the assessment of DV cases, frequently considering reporting situations where the violence is perceived as more serious and potentially life threatening. This is the approach recommended by the Portuguese General Health Department [ 21 ]. The focus on the degree of violence and the position assumed by family doctors in Portugal when faced with more extreme cases, with possible life-threatening consequences, may prevent some tragic conclusions, however, it may be considered insufficient in the majority of cases. Qualitative studies conducted in the Philippines and Peru suggest that women consider psychological abuse more degrading and intolerable than physical violence and the majority of victims of DV seek medical help more frequently as a result of prolonged cycles of intimidation than as a response to an isolated episode of abuse [ 22 ]. The threat of more serious forms of violence can be sufficient to compromise the health of the victim. A USA study conducted in Texas investigating the relationship between exposure to firearms and negative health outcomes on DV victims showed that the ownership of a firearm by the aggressor is significantly associated with worse victim’s physical health even in cases where firearm related DV never occurred [ 23 ].

Our study focused on the opinions of family doctors regarding mandatory reporting, and what compels them to present or not a report. The success of mandatory reporting laws is linked with the degree of compliance by health care professionals with the legislation. However, studies on the efficiency of mandatory reporting are limited. A USA study based on data from the Los Angeles Sheriff’s Department analysed the number of dispatches for DV in the years prior and after the implementation of mandatory reporting legislation, observing no differences in the number of reports by health care professionals [ 24 ]. This was attributed to many reasons: lack of knowledge about the new law, lack of compliance, and deterrence from the victim in divulging the abuse. The barriers and facilitators encountered in or study could provide an additional explanation to the degree of compliance by health care professionals with the law.

The sparsity of studies analysing the efficiency of mandatory reporting laws of DV and the motives that lead health professionals to report, or not, these cases to the authorities, prevents a complete understanding of this kind of legislation and its impact on the lives of victims, aggressors, and individuals faced with the obligation to report.

Implication of the findings for future practice and research

Our study demonstrates the need to intervene with family doctors in Portugal to improve their response to DV cases. Importantly, most of the barriers found in our study are related to the physician’s activity. This calls for the creation of educational programs developed to answer the barriers encountered and faced by family doctors from detection until reporting DV cases. More training opportunities should be available, and doctors should be allowed to manage their consultations more freely enabling them to allocate more time to oversee not only DV cases but other social issues.

In 2014, the General Directorate for Health (DGS) has published a document addressing the approach, diagnoses and intervention of interpersonal violence by health services [ 21 ]. This document needs to be further disseminated among health professionals and can serve as a guide for a more focused and community base intervention. Since primary care in Portugal is organized in regional-based unities, current guidelines should be revalued, and protocols established based on the resources available in each community, namely the accessibility of specialized services, support institutions, and police and juridical services. Each health unit should possess a comprehensive and easy to follow algorithm to facilitate the management and reporting of DV cases. An investment should be made to provide support to organizations, and specifically EPVA’s, groups formed in each health unit to respond to DV, with greater financial and human resources.

The DGS reported an increase in violence against health professionals of 40% in 2022 when compared with the previous year [ 25 ]. Resources should be allocated to guarantee health professionals’ security, allowing them to conduct their practice in a safe environment, without fear of retaliation when faced with a DV aggressor.

The apparent conflict between mandatory reporting and doctors-patient confidentiality should also be addressed. According to the DGS doctors-patient confidentiality should only be broken in extreme cases of violence that put at risk the imminent security of the victim. However, the evaluation of the degree of violence is based on the understanding of the doctor of each situation. This approach can be highly subjective, compromising health care services efficiency in responding to DV cases, especially when associated with the lack of training and knowledge cited by the doctors.

To establish an effective response to DV, it is also necessary to understand the phenomenon in all its dimensions. Further research through qualitative studies with both victims and aggressors should be conducted, to investigate their perceptions of their interactions with different services: the health care system, support institutions, law enforcement, and the juridical system. Research that evaluates the experiences and opinions of other professionals, namely doctors with other medical specialities and across diverse clinical sectors, nurses, and law enforcement, should also be considered.

The efficiency of mandatory reporting laws in providing victims protection and persecuting aggressors should be carefully assessed. It is not enough to make the reporting of DV cases mandatory. The success of mandatory reporting laws and the standard of compliance with this kind of legislation depends on the perception of the degree of support and protection offered to the victims. Interventions that focus on the social and juridical responses to DV should be implemented to guaranty the success of the legislation. If, however, after careful study and consideration, mandatory reporting laws fail to provide an efficient and protective response to DV, this legislation should be reconsidered.

This study investigated the motivations of family doctors in Portugal to present, or not, a report of DV cases to the authorities, and adds knowledge to an under researched subject providing the bases for a wider debate concerning current practices and legislation. The themes and subthemes identified in this study represent several of the international concerns and opinions voiced by healthcare professionals in other parts of the world.

The mitigation and prevention of DV imply a continuous effort on a social and legislative level to answer victims’ necessities efficiently and protectively. The results of this study can help develop new interventions for DV, and the development of new clinical, social and legislative approaches.

Availability of data and materials

The anonymised datasets analysed during the current study are not publicly available due to concerns regarding participants’ confidentiality but are available from the corresponding author upon reasonable request.

Abbreviations

Portuguese Support Victims Association

Domestic Violence

Adult Violence Preventions Teams

Republican National Guard

Public Security Police

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Acknowledgements

We would like to thank the support received in the Master on Legal Medicine at the Institute of Biomedical Sciences Abel Salazar (ICBAS) at the University of Porto.

This research did not receive any specific grant for funding.

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Diana Nadine Moreira & Mariana Pinto da Costa

Institute of Public Health of the University of Porto, Porto, Portugal

Mariana Pinto da Costa

Institute of Psychiatry, Psychology & Neuroscience, King’s College London, 16 De Crespigny Park, London, SE5 8AB, UK

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This article reports work from the Masters of DNM under the supervision of MPC. DNM and MPC contributed equally to writing this article.

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Correspondence to Mariana Pinto da Costa .

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Moreira, D.N., Pinto da Costa, M. Barriers and facilitators of the reporting by family doctors of cases of domestic violence – a qualitative study across Portugal. BMC Prim. Care 25 , 107 (2024). https://doi.org/10.1186/s12875-024-02329-0

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Expression analysis of zinc-metabolizing enzymes in the saliva as a new method of evaluating zinc content in the body: two case reports and a review of the literature

• Ken-ichiro Sakata 1 ,
• Ayako Hashimoto 2 ,
• Taiho Kambe 3 ,
• Jun Sato 1 ,
• Noritaka Ohga 1 ,
• Yutaka Yamazaki 4 ,
• Masahide Koyachi 5 ,
• Itagaki Tatsuki 1 ,
• Mai Okada 1 ,
• Okura Taro 1 ,
• Hiroshi Hikasa 1 &
• Yoshimasa Kitagawa 1  

Journal of Medical Case Reports volume  18 , Article number:  198 ( 2024 ) Cite this article

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The activity level of alkaline phosphatase, a zinc-requiring enzyme in the serum, is used to indicate zinc nutritional status; however, it does not correlate with serum zinc levels or subjective symptoms of taste disorder in many cases. Hence, this study focused on the total activity of alkaline phosphatase, a zinc-requiring enzyme. The total alkaline phosphatasa activity level in the saliva was measured before and after zinc supplementation, and the results were compared with serum zinc levels.

Case presentation

This study included patients with hypozincemia, specifically a patient with zinc-deficient taste disorder (patient 1: a 69-year-old Japanese woman) and a patient with glossodynia with zinc deficiency (patient 2: an 82-year-old Japanese woman). Saliva samples were collected, and blood tests were performed before and after zinc supplementation. Subjective symptoms and serum zinc levels were simultaneously evaluated. Zinc supplementation was performed using zinc acetate hydrate or Polaprezinc.

Conclusions

Total alkaline phosphatase activity levels were found to be associated with serum zinc levels and subjective symptoms. A further study with a higher number of patients is necessary to confirm whether total alkaline phosphatase activity levels more accurately reflect the amounts of zinc in the body than serum zinc levels.

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Zinc deficiency in humans was recognized for the first time in 1961 [ 1 ]. Studies have reported that zinc deficiency is associated with taste disorder and glossodynia [ 2 , 3 , 4 ]. There are several other symptoms, such as bedsores, skin symptoms, delayed wound healing, fetal growth restriction, chronic diarrhea, anemia, and altered mental status [ 5 ]. The Zincage Project (research on the relationship between health/aging and zinc), an epidemiological survey in Europe, has shown that health status of older adults is positively associated with the amount of zinc in the body [ 6 ]. In the basic research field, zinc is considered an essential trace metal element, with approximately 2 g being present in the body. Zinc plays a role as the active center of 3000 types of enzymes and bioactive substances [ 7 ]. Recent human genome decoding projects have indicated that approximately 10% of genes have zinc-binding domains. Hence, researchers have focused their attention on the relationship between zinc and diverse physiological functions, including zinc transporters and zinc signals [ 8 ]. Sequelae of oral diseases caused by zinc deficiency lead to a frailty cycle. This cycle stems from appetite loss and decreased food consumption, causing undernutrition. This state results in weight loss and sarcopenia onset (Fig.  1 ).

Hypozincemia leads to frailty

To address this issue, accurate measurement of the zinc content in the body is required in clinical practice, highlighting the need to develop a method for accurate and rapid diagnosis of zinc deficiency. Zinc deficiency is known as the main factor contributing to taste disorder [ 9 ]. However, it is not well known that zinc deficiency can also cause several oral diseases, such as mouth ulcers and glossodynia, posing a diagnostic and treatment challenge in many cases [ 10 ]. Serum zinc levels are a commonly used indicator of zinc assessment in clinical practice. However, they do not accurately reflect the zinc content in the body, because they account for only less than 0.1% of the total zinc in the body [ 3 ]. Furthermore, they exhibit circadian rhythms and are easily influenced by several conditions. Owing to such disadvantages, they are not considered an absolute indicator [ 11 ]. In this study, we attempted to identify an indicator other than serum zinc levels for accurately measuring the amount of zinc in the body, focusing our attention on zinc-requiring enzymes, which show zinc-dependent activity. Zinc-requiring enzymes are activated after binding to zinc. The activity level of alkaline phosphatase (ALP), a zinc-requiring enzyme in the serum, is used as an indicator of zinc nutritional status; however, it does not correlate with serum zinc levels or subjective symptoms of taste disorder in many cases. To accurately evaluate the zinc nutritional status, we shifted our focus from serum ALP to total alkaline phosphatase [ 12 , 13 ]. Four ALP isozymes are  broadly expressed in various cells (Table  1 ). Previous studies using cultured cells and animals have shown that ALP, an enzyme that is activated by coordinating zinc at the active center, plays a role in suppressing inflammation by hydrolyzing adenosine triphosphate (ATP), which serves as a danger signal extracellularly, into adenosine. The results demonstrate that ALP is a zinc-requiring enzyme (zinc marker enzyme), enabling the accurate measurement of zinc content in the body [ 14 ].

Objective: Intervention is required to break the frailty cycle; however, the following questions remain unsolved: “Which indicators should be used for zinc supplementation?”, “How long should zinc supplementation be continued?”, and “How much zinc is required for the supplementation?” We raised the possibility of conducting translational research in which basic scientific findings are translated into clinical practice, with the aim of attaining a society where “older adults can enjoy meals.” This study aimed to evaluate total ALP activity levels in the saliva, serum zinc levels, and subjective symptoms before and after zinc supplementation in patients with oral diseases caused by zinc deficiency.

Patients and methods

Saliva samples were used because they can be collected repeatedly in a noninvasive manner. They were collected before and after zinc supplementation, and subjective symptoms of taste disorder and serum zinc levels were simultaneously evaluated. Saliva sample collection and blood tests were performed only in the morning. The gustatory testing (whole mouth) results are as follows: Four types of taste solutions [sucrose (S), sodium chloride (NaCl; N), tartaric acid (T), and quinine hydrochloride dihydrate (Q)] were dropped on the dorsum of the tongue at the following concentrations (No. 1: 2.9 μmol, No. 1.5: 5.8 μmol, No. 2: 8.8 μmol, No. 2.5: 17.5 μmol, No. 3: 29.2 μmol, No. 4: 58.4 μmol, and No. 5: 2337 μmol), and cognitive thresholds were tested by tasting the samples within 2–3 seconds. Taste perception threshold testing followed the criteria in the table below, and when No. 5 could not be recognized, it was considered taste loss. The efficacy of zinc supplementation in patients with taste disorder and those with burning mouth syndrome (BMS) was evaluated using the Visual Analog Scale [ 15 ] and Numerical Rating Scale [ 16 ], respectively. Zinc content was measured using ACCURAS AUTO Zn (Shino-Test Corporation, Osaka, Japan).

Zinc was measured using the Acurus Auto Zn (Shino-Test Corporation, Tokyo, Japan) based on the colorimetric method using an automated analyzer (Hitachi 7700 series; Hitachi High-technology Corporation, Tokyo, Japan). Measurement of ALP activity in saliva was performed as follows: 5 µl of saliva was used to measure ALP activity. A total of 100 μl of substrate solution [2 mg/ml disodium  p -nitrophenylphosphate hexahydrate ( p NPP); Wako Pure Chemicals in 1 M diethanolamine buffer pH 9.8 containing 0.5 mM MgCl 2 ] was added and incubated at 37 ℃ for 1 hour. The released  p -nitrophenol product was quantified by measuring the absorbance at 405 nm.

Calf intestinal ALP (Promega) was used to generate a standard curve. This study was approved by the Hokkaido University Hospital Independent Clinical Research Review Committee (Approval No. 019-0337) and approved by the Ethics Committee of Kyoto, University Graduate School and Faculty of Medicine (Approval No. R3275).

All study procedures were performed in accordance with the principles of the Declaration of Helsinki. Written informed consent was obtained from all participants.

Two patients experiencing hypozincemia were included. Patient 1 was a 69-year-old Japanese woman (Fig.  2 ). The patient presented to the Dentistry Center, Hokkaido University Hospital, with the chief complaint of hypogeusia. In addition to routine intraoral examination, the patient received gustatory testing, oral bacteria testing, blood tests, and chewing gum test. Intraoral examination showed mild xerostomia and tongue coating. The results of gustatory testing (whole mouth) were sweet (2.5), salty (4), sour (2), and bitter (2), indicating hypogeusia. Oral bacteria testing was unremarkable. The volume of saliva collected using the chewing gum test was 17.8 mL. Blood tests at the initial visit showed a serum zinc level of 65.6 μg/dL. Based on this low serum zinc level, the patient was diagnosed with zinc-deficient taste disorder. The total ALP activity level was 0.072 mU/5 μL. Zinc supplementation was continued for 2 months using zinc acetate hydrate. After the patient orally took zinc acetate hydrate 50 mg for 1 month, the serum zinc level improved to 115.3 μg/dL, and the total ALP activity level increased to 0.102 mU/5 μL. After the zinc acetate hydrate dose was reduced to 25 mg, the serum zinc level slightly decreased to 86 μg/dL. However, the total ALP activity level remained almost the same at 0.1 mU/5 μL.

69-year-old woman with taste disorder

Patient 2 was an 82-year-old Japanese woman (Fig.  3 ). The patient presented to the Dentistry Center, Hokkaido University Hospital, with the chief complaint of glossodynia. In addition to routine intraoral examination, she received gustatory testing, oral bacteria testing, and blood tests. Intraoral examination showed mild tongue coating. The results of gustatory testing (whole mouth) were sweet (1.5), salty (2.5), sour (2.5), and bitter (2), indicating hypogeusia. Results of oral bacteria testing were positive for Candida albicans . Administration of amphotericin B gargles eradicated C. albicans . However, glossodynia persisted after the eradication; therefore, the patient was diagnosed with BMS secondary to zinc deficiency. Blood tests at the initial visit showed a serum zinc level of 51.9 μg/dL. On the basis of this low serum zinc level, the patient was diagnosed with BMS secondary to zinc deficiency. The total ALP activity level was 0.045 mU/5 μL. Zinc supplementation was continued for 5 months using zinc acetate hydrate. After the patient orally took zinc acetate hydrate 50 mg for 1 month, the serum zinc level improved to 113.3 μg/dL, and the total ALP activity level increased to 0.05 mU/5 μL. The patient received a reduced zinc acetate hydrate dose of 25 mg for the subsequent month of zinc supplementation, and the serum zinc level and total ALP activity level 2 months after treatment initiation were 126.6 μg/dL and 0.03 mU/5 μL, respectively. Zinc acetate hydrate 25 mg was administered every other day over the third and fourth months after the initiation of zinc supplementation. Serum zinc and total ALP activity levels 4 months after treatment initiation were 71.3 μg/dL and 0.049 mU/5 μL, respectively. Zinc acetate hydrate 50 mg was administered for the fifth month after the initiation of zinc supplementation. Serum zinc and total ALP activity level 5 months after treatment initiation were 116.6 μg/dL and 0.056 mU/5 μL, respectively. The serum zinc level decreased transiently, but the total ALP activity level remained almost the same.

An 82-year-old woman with glossodynia

Discussion and conclusions

Unstable serum zinc levels.

Currently, zinc supplementation is performed on patients with taste disorder and glossodynia due to zinc deficiency based on serum zinc levels. Our previous study showed that the mean serum zinc level was 72 μg/dL in the taste disorder group and 74 μg/dL in the control group, with no relationship between taste disorder and serum zinc levels [ 17 ]. The proportion of patients with zinc deficiency in the taste disorder group was significantly higher ( p  < 0.05) only when a serum zinc level of < 60 μg/dL (that is, severe zinc deficiency) was used to identify patients with zinc deficiency. Possible explanations for the absence of an association between clinical symptoms and serum zinc levels include: (1) Serum zinc levels do not accurately reflect the zinc content in the body; (2) serum zinc levels exhibit a large fluctuation of circadian rhythms and are easily influenced by meals, stress, and hormone conditions, therefore making them problematic as an indicator of zinc deficiency; and (3) differences in serum zinc levels between laboratories (serum zinc levels tend to show a higher value in outsourcing examination than in-hospital examination owing to hemolysis). These disadvantages suggest that serum zinc levels may not reflect the bioavailability of zinc in the body in some cases. Thus, selecting an appropriate sample and developing a method to analyze the sample are important issues.

Taste disorder and zinc

The relationship between taste disorder and zinc deficiency plays an important role in regenerating taste bud cells. Taste buds, or taste sensors, are actively regenerated approximately every 10 days. Normal regeneration of taste bud cells is required to correctly recognize different tastes. Insufficient regeneration of taste bud cells due to zinc deficiency is associated with a decrease in the number of these cells, causing impairment of taste sensor function. Zinc plays an important role in protein formation that is required for cell regeneration and transmission of genetic information.

Glossodynia and zinc

Glossodynia is a traditionally well-known symptom of hypozincemia in clinical practice; however, its evident cause is unknown [ 18 ]. Zinc exerts analgesic effects when administered locally to the spinal nerves or peripheral nerves or systemically in patients with chronic pain in areas other than in the oral area, renal dysfunction, anemia, and itchy sensation on the skin [ 19 , 20 ]. The mechanism of this analgesic effect was reported in a study in 2011 [ 21 ]. In the said study, increased sensitivity to pain was observed in knockout mice whose zinc ion binding to the NMDA-type glutamate receptor was inactivated. The authors reported that C fibers were particularly affected as compared with Aδ fibers, resulting in difficulty in suppressing chronic pain. Chronic glossodynia signals are transmitted to the brain mainly via C fibers, suggesting that the association between hypozincemia and glossodynia may be mediated by NMDA receptors, although the details remain to be clarified.

Total ALP activity levels

Both patients experienced a transient decrease in serum zinc levels, but total ALP activity levels did not change. Total ALP activity is involved in the dephosphorylation of various physiological substrates and has vital physiological functions, including extraskeletal functions such as neuronal development, detoxification of lipopolysaccharide, an anti-inflammatory role, bile pH regulation, and maintenance of the blood–brain barrier. Total ALP activity has also been reported to be involved in cardiovascular calcification, chronic kidney disease, and type 2 diabetes [ 22 ]. In particular, it is a logical clinical target to attenuate vascular calcification. Total ALP activity has gained attention as a clinical marker in several diseases. The relationship between total ALP activity levels and changes in zinc content in the body has been evaluated only in studies using cultured cells and animals. Thus, this is the first study to report the relationship in humans. Furthermore, this study used saliva samples; therefore, the results are extremely useful because these samples can be collected repeatedly in a noninvasive manner. Further cross-sectional or cohort studies are required to clarify the relationship between serum zinc levels and zinc-requiring enzymes, including total ALP activity, to interrupt the disease cycle due to zinc deficiency and improve the health status. This study suggests that total ALP activity may be a new diagnostic marker alternative to serum zinc levels. Furthermore, it may be used as an indicator when assessing the efficacy of zinc supplementation.

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No new data were created or analyzed in this study. Data sharing is not applicable to this article.

Abbreviations

Alkaline phosphatase

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Acknowledgements

I would like to give my heartfelt thanks to Prof. Kitagawa whose comments and suggestions were immeasurably valuable throughout the course of my study. I am also in debt to Prof. Yamazaki whose meticulous comments were enormously helpful. I would like to express my gratitude to Mr. Taiho Kambe and Ayako Hashimoto, for the valuable advice for evaluating zinc content in the body. I would also like to thank my family for their moral support and warm encouragement.

This study was partially supported by Lotte Co., Ltd., Tokyo, Japan.

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Department of Oral Diagnosis and Medicine, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan

Ken-ichiro Sakata, Jun Sato, Noritaka Ohga, Itagaki Tatsuki, Mai Okada, Okura Taro, Hiroshi Hikasa & Yoshimasa Kitagawa

Department of Food and Nutrition, Faculty of Home Economics, Kyoto Women’s University, Kyoto, Japan

Ayako Hashimoto

Department of Applied Molecular Biology, Division of Integrated Life Science, Graduate School of Biostudies, Kyoto University, Kyoto, Japan

Taiho Kambe

Department of Gerodontology, Division of Oral Health Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan

Yutaka Yamazaki

Department of Oral Pathobiological Science and Surgery, Tokyo Dental College, Tokyo, Japan

Masahide Koyachi

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Contributions

Conceptualization: KS and JS. Methodology/formal analysis: AH and TK. Investigation: OT and TI. Resources: KS and NP. Data curation: KS. Writing—original draft preparation: KS. Writing—review and editing: YY. Visualization: NO, MO, and HH. Supervision: YK. Project administration: KM. All authors have read and agreed to the published version of the manuscript.

Corresponding author

Correspondence to Ken-ichiro Sakata .

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Ethics approval and consent to participate.

In this report, we anonymized personal information and ensured the protection of privacy. This study was approved by the Hokkaido University Hospital Independent Clinical Research Review Committee (Approval No. 019–0337) and approved by the Ethics Committee of Kyoto, University Graduate School and Faculty of Medicine (Approval No. R3275). Written informed consent was obtained from all participants.

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Written informed consent was obtained from the patients for  publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

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The authors declare no competing interests.

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Sakata, Ki., Hashimoto, A., Kambe, T. et al. Expression analysis of zinc-metabolizing enzymes in the saliva as a new method of evaluating zinc content in the body: two case reports and a review of the literature. J Med Case Reports 18 , 198 (2024). https://doi.org/10.1186/s13256-024-04463-w

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Pathological findings with vacuoles in anti-mitochondrial antibody-positive inflammatory myopathy

• Yuanchong Chen 1 , 2 ,
• Wei Zhang 1 ,
• Zhaoxia Wang 1 ,
• Hongjun Hao 1 ,
• Yun Yuan 1 &
• Yiming Zheng 1  

BMC Musculoskeletal Disorders volume  25 , Article number:  257 ( 2024 ) Cite this article

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A few patients with inflammatory myopathy showed anti-mitochondrial antibody (AMA) positivity. This study aimed to report the clinical and pathological findings with vacuoles in 3 cases of such patients.

Three cases with myositis from the Myositis Clinical Database of Peking University First Hospital were identified with AMA positivity. Their clinical records were retrospectively reviewed and the data was extracted. All the 3 cases underwent muscle biopsy.

Three middle-aged patients presented with chronic-onset weakness of proximal limbs, marked elevation of creatine kinase, and AMA-positivity. Two of the 3 cases meet the criteria of primary biliary cholangitis. All the 3 cases presented with cardiac involvement and proteinuria. Two cases developed type 2 respiratory failure. MRI of the thigh muscle showed multiple patches of edema bilaterally in both cases, mostly in the adductor magnus. Pathological findings include degeneration of muscle fibers, diffused MHC-I positivity, and complement deposits on cell membranes. Vacuoles without rims of different sizes were discovered under the membrane of the muscle fibers. A few RBFs were discovered in case 1, while a diffused proliferation of endomysium and perimysium was shown in case 2.

Conclusions

AMA-positive inflammatory myopathy is a disease that could affect multiple systems. Apart from inflammatory changes, the pathological findings of muscle can also present vacuoles.

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Anti-mitochondrial antibody (AMA), a specific autoantibody of primary biliary cholangitis (PBC), can also be associated with inflammatory myopathy [ 1 ]. AMA is not considered to be associated with disease severity [ 2 ], and it has not been classified as myositis-specific autoantibodies (MSAs) or myositis-associated autoantibodies (MAAs) yet. First reported in 1974 [ 3 ], this type of inflammatory myopathy is still not well recognized. The prevalence of AMA positivity is around 5–10% in patients diagnosed with myositis [ 1 , 4 ]. Cardiac involvement and pulmonary hypertension were reportedly associated with AMA-positive myositis [ 5 ]. On biopsy, there showed only non-specific inflammatory features of myositis [ 4 ].

To date, the clinical, histopathological, and radiological features of AMA-positive myositis have not been clarified. In this study, we reported 3 cases of AMA-positive myositis with various types of cardiac involvement, renal involvement, and respiratory failure. A newly discovered pathological finding of vacuole formation is also presented in all 3 cases. Their radiological findings were also analyzed in this study.

Patients and clinical data

After being approved by the local institutional review board, medical records of those patients who were positive for AMA and diagnosed with myositis were retrospectively reviewed. Informed consent from the included cases was obtained according to the approved study protocol. The results of clinical evaluation including symptoms, duration of muscle weakness, and Medical Research Council (MRC) scale for muscle strength were extracted from the medical records. The presence of AMA was tested by indirect immunofluorescence, and AMA-M2 was titrated by enzyme-linked immunosorbent assay. Muscle MRI examinations of both thighs were performed in patients using a 1.5T MR scanner (GE 1.5T Signa Twin Speed, GE Healthcare; Multiva 1.5T, Philips Healthcare) with axial scanning in conventional T1-weighted imaging (T1WI) and short time inversion recovery (STIR) sequences. The top part of the coil was at the level of the anterior superior iliac spine, and the scanning range covered from the pelvis to the thigh. The patients were asked to rest for at least 30 min before imaging to avoid the effects of activity or exercise. The muscles were scanned in a noncontracted state. The total acquisition time for the axial T1-weighted and STIR images was approximately 20 min.

Muscle biopsy

With written informed consent, the muscle biopsy was conducted. Serial frozen sections were obtained, and stained using hematoxylin and eosin (H&E), modified Gomori trichrome (mGT), periodic acidic Schiff (PAS), oil red O, adenosine triphosphatase (ATPase) at pH 10.6, 10.5, 4.5 and 4.6, NADH dehydrogenase, succinate dehydrogenase (SDH), cytochrome c oxidase (COX), and nonspecific esterase (NSE) according to standard procedures. Sections were observed under a light microscope.

Clinical findings

Three middle-aged patients presented with chronic-onset weakness of proximal limbs, marked elevation of creatine kinase, and AMA-positivity, accompanied by proteinuria (3 cases), cardiac involvement (2 cases), respiratory failure (2 cases) as well as other autoimmune diseases (primary biliary cholangitis – 3 cases, autoimmune hepatitis – 1 case, Hashimoto thyroiditis – 1 case). Note that none of the cases showed positivity of MSAs, including anti-melanoma differentiation-associated gene 5 (MDA5), anti-small ubiquitin-like modifier activating enzyme 1 (SAE1), anti-Mi2, anti-transcription intermediary factor 1γ (TIF1γ), anti-nuclear matrix protein 2 (NXP2), anti-signal recognition particle (SRP), and anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (HMGCR) autoantibodies. The clinical characteristics of the three cases were shown in Table  1 . Details of clinical findings as well as treatment responses were described in Supplementary Material .

Two cases of this study underwent MRI except for case 3 because of an implanted MRI-incompatible pacemaker. In case 1, the muscle MRI of the thigh showed multiple regions of hyperintensity on STIR sequences, especially in the quadriceps femoris and adductor magnus. The thigh MRI of case 2 showed hyperintensities of the vastus medialis and adductor magnus bilaterally on STIR, indicating edema of muscle. Additionally, case 2 had hyperintensities on diffusion weighted imaging (DWI) (b-value = 800 s/mm 2 ). Fatty degeneration was not significant in these 2 cases (Fig.  1 ).

Right thigh MRI of 2 cases. Axial T1WI ( A ) and STIR ( B ) images of case 1, and axial T1WI ( C ) and STIR ( D ) images of case 2were shown

Pathological findings

Muscle biopsy of case 1 was obtained from the left deltoid. Histological findings of biopsy specimens showed necrosis and regeneration of muscle fibers. MHC-I was diffusely expressed, and several non-necrotic fibers had complements deposits on their membranes. Vacuoles in different sizes were discovered under the membrane of the muscle fibers. A biopsy on the right tibialis anterior of case 2 showed diffused proliferation of endomysium and perimysium with a characteristic ‘checkerboard’ pattern of the muscle fascicles and no inflammatory cell infiltrate, accompanied by regeneration and vacuolization of a few muscle fibers. In case 3, muscle biopsy was obtained from the right biceps brachii. Histological findings showed many necrotic and regenerating muscle fibers. MHC-1 positivity, complement deposits, and a few vacuoles in fibers were noticed. Pathological findings of case 1 and 3 were consistent with immune-mediated necrotizing myopathy (IMNM), while case 2 showed the pattern of proliferative myositis. The vacuoles of these patients are mostly located under the membrane, multiple, without rims, with different sizes, and are not stained in ORO, PAS, NSE and NADH staining. (Fig.  2 )

Muscle histopathological findings of case 1 ( A, B, C, D ), case 2 ( E, F, G, H ), and case 3 ( I, J, K, L ). Low-power field of hematoxylin and eosin (H&E) staining ( A, E, I ), high-power field of H&E staining ( B, F, J ), modified Gomori trichrome (mGT) staining ( C, G, K ), and nicotinamide adenine dinucleotide (NADH) staining ( D, H, L ) were shown

The immunohistochemical staining results are showed in Supplementary Fig.  1 and Supplementary Table  1 .

The association between myositis and AMA-positivity had been reported previously. In the presence of AMA-positivity, the subtypes of myositis include polymyositis, necrotizing autoimmune myopathy, and dermatomyositis [ 4 ]. Common clinical features include muscle weakness (torso or limb) and cardiac involvement. Rare manifestations, such as pulmonary hypertension and renal involvement, were also presented in this study. Some studies showed a lower chance of limb weakness in AMA-positive myositis, compared to AMA-negative ones [ 6 ]. Paravertebral muscle atrophy presented by head drop [ 7 ] or rectus abdominis involvement without weakness [ 8 ] may be a clinical feature of AMA-positive myositis. This may indicate torso muscle biopsy, other than biceps brachii or quadriceps femoris, to confirm the diagnosis. However, in this study, limb weakness was presented in all these 3 cases and torso weakness was presented in none of the cases. All the 3 cases met the criteria of myositis and none of them showed the characteristic skin rash of dermatomyositis.

Cardiac involvement can be present accompanied by AMA-positive myositis, or only AMA positivity without skeletal muscle diseases. In patients with idiopathic inflammatory myopathy, AMA-M2 positivity presented more frequently in those who had myocarditis than in those who did not. Patients with AMA-M2 positivity were more likely to have decreased left ventricular ejection fraction, right ventricular ejection fraction, and a higher chance of right atrial enlargement [ 9 ]. The manifestations include atrial fibrillation, atrioventricular block, complete right bundle branch block (case 1), premature ventricular contraction (case 2, 3), and ventricular tachycardia [ 10 ]. Supraventricular arrhythmias showed a higher prevalence in patients with AMA-positive myositis that suggesting atrial myocardial involvement [ 1 , 11 ]. Dilated cardiomyopathy (DCM) of case 2 may be related to AMA positivity as reported [ 12 , 13 ], but the BAG3 mutation may also cause DCM. Further workup may be needed to confirm the root cause of DCM.

Pulmonary hypertension, as presented in case 3, can also be associated with AMA-positive myositis. As reported in a case report, mean pulmonary artery pressure (PAP) could rise to 28 mmHg (measured by right heart catheterization). Pulmonary hypertension might be a result of left-sided heart failure caused by cardiomyopathy. The reduced ejection fraction of 41.9% and marked increased PAP of 44.2 mmHg (measured by echocardiogram) were evident in case 3. This may contribute to the process of type 2 respiratory failure presented in both case 2 and case 3, the most significant feature of AMA-positive myositis [ 14 ], in addition to respiratory muscle weakness.

Renal involvement of myositis was often considered a result of tubal blockage by myoglobin released from damaged muscle cells, leading to post-renal acute kidney injury. Long-lasting duration would accelerate the process toward chronic kidney disease [ 15 , 16 ]. This kind of renal involvement can be monitored by serum creatinine. Another mechanism of renal involvement may be caused by an autoimmune response, such as complement activation in dermatomyositis [ 17 ] or overlapping connective tissue diseases that may be present in case 2 as the level of complement C4 was decreased. PBC, a disease characterized by AMA positivity, could be reportedly associated with multiple myeloma [ 18 , 19 , 20 , 21 , 22 ] or systemic lupus erythematosus [ 23 , 24 , 25 , 26 ] that leads to proteinuria. It has not been reported that AMA-positive myositis or PBC could present with proteinuria in the absence of other autoimmune diseases. In this study, all 3 cases presented with proteinuria without elevation of serum creatinine. No significant evidence showed overlapping connective tissue disease. Further workup like renal biopsy may be needed to undercover the mechanism of renal involvement of these patients.

The characteristic imaging pattern of AMA-positive myositis had been reportedly concluded as a “cuneiform sign” (hyperintensities mostly in adductor magnus on STIR) as well as fatty degeneration of adductor magnus and semimembranosus [ 27 ]. In 2 cases of this study who underwent thigh MRI, both of them presented with hyperintensities in the adductor magnus on STIR, but only case 2 had mild fatty degeneration of muscles. Hyperintensities in quadriceps femoris were also presented in these 2 cases. Case 2 also has hyperintensities on DWI that may indicate the cytotoxic mechanism of muscle edema. This can be inferred by mitochondrial injury due to the immune response induced by AMA. Muscles including adductor magnus and quadriceps femoris are more likely to be involved because of their rather high content of mitochondria [ 27 , 28 ].

Pathological findings of AMA-positive myositis have not been comprehensively reported thus far. Studies showed that infiltration of inflammatory cells, endomysium proliferation, fiber necrosis, and regeneration as well as diffused expression of MHC-II may present in muscle biopsy [ 4 , 8 , 10 , 27 ]. Two and one cases in the present study showed the pattern of IMNM or proliferative myositis which has not been reported before, respectively. However, the specific pathological findings of AMA-positive myositis have not been thoroughly described. This study also showed non-rimmed vacuolization of muscle fibers which may be a new characteristic of AMA-positive myositis that has not been discovered recently [ 4 , 5 , 8 , 10 , 29 , 30 ]. The vacuole in muscle fibers is a non-specific myopathological change, occurring in several rare disorders. Rimmed vacuoles were thought to be the accumulation of autophagic vacuoles caused by lysosomal dysfunction or disruption of the autophagic process [ 31 , 32 ], reported in one case of AMA-positive myositis [ 29 ]. Unlike rimmed vacuoles, the formation of non-autophagic vacuoles involves vacuolation of myofibril endocellular organelles and local accumulation of metabolic substances, mainly in muscle diseases with glucose and lipid metabolism disorders, periodic paralysis, and other genetically heterogeneous diseases [ 33 , 34 ]. All the 3 cases in this study showed vacuoles, located just underneath the membrane, or intracellularly, without glucose and lipid storage. The mechanism of vacuole formation in these patients with AMA-positive myositis may be different from rimmed vacuoles or glucose and lipid storage, and further studies are needed to confirm it, especially some ultrastructural studies. The occurrence rate and the characteristics of non-rimmed vacuoles among different types of inflammatory myopathies are also to be unveiled in the future.

There are limitations to this study. The present study reported a limited number of cases because of the rareness of AMA-positive myositis. The clinical manifestations, imaging patterns, and pathological findings were reported, but the underlying mechanism and correlation remained unclear. Further studies were needed to investigate the mechanism of vacuole formation and the percentage of muscle fiber vacuolization in AMA-positive myositis in comparison with other types of inflammatory myopathy.

AMA-positive myositis is a disease that could cause multi-system involvement, including respiratory, circulation, and musculoskeletal systems. On thigh MRI, adductor magnus and quadriceps femoris could present with edema. Vacuoles without rims that differed from inclusion body myositis may appear on pathological examination.

Data availability

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

Abbreviations

• Anti-mitochondrial antibody

Primary biliary cholangitis

Myositis-specific autoantibodies

Myositis-associated autoantibodies

Melanoma differentiation-associated gene 5

Small ubiquitin-like modifier activating enzyme 1

Transcription intermediary factor 1γ

Nuclear matrix protein 2

Signal recognition particle

3-hydroxy-3-methylglutaryl-coenzyme A reductase

Medical Research Council

T1-weighted imaging

Short time inversion recovery

Hematoxylin and eosin

Modified Gomori trichrome

Periodic acidic Schiff

Adenosine triphosphatase

Succinate dehydrogenase

Cytochrome c oxidase

Nonspecific esterase

Diffusion weighted imaging

Immune-mediated necrotizing myopathy

Dilated cardiomyopathy

Pulmonary artery pressure

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Acknowledgements

The authors extend their appreciation to the patients for their support. The authors also acknowledge the entire staff of the Department of Neurology, Peking University First Hospital.

This study was supported by the National Natural Science Foundation of China (Grant No. 82201550).

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Yuanchong Chen, Wei Zhang, He Lv, Zhaoxia Wang, Hongjun Hao, Yun Yuan & Yiming Zheng

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YC contributed to the drafting of the manuscript. WZ, HL, ZW and YY contributed to revision of the manuscript. YZ contributed to the concept and revision of the manuscript. The authors read and approved the final manuscript. All authors met the authorship criteria from the International Committee of Medical Journal Editors (ICMJE).

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Chen, Y., Zhang, W., Lv, H. et al. Pathological findings with vacuoles in anti-mitochondrial antibody-positive inflammatory myopathy. BMC Musculoskelet Disord 25 , 257 (2024). https://doi.org/10.1186/s12891-023-06941-6

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Received : 28 February 2023

Accepted : 06 October 2023

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DOI : https://doi.org/10.1186/s12891-023-06941-6

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