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The case study approach

• Sarah Crowe 1 ,
• Kathrin Cresswell 2 ,
• Ann Robertson 2 ,
• Guro Huby 3 ,
• Anthony Avery 1 &
• Aziz Sheikh 2  

BMC Medical Research Methodology volume  11 , Article number:  100 ( 2011 ) Cite this article

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The case study approach allows in-depth, multi-faceted explorations of complex issues in their real-life settings. The value of the case study approach is well recognised in the fields of business, law and policy, but somewhat less so in health services research. Based on our experiences of conducting several health-related case studies, we reflect on the different types of case study design, the specific research questions this approach can help answer, the data sources that tend to be used, and the particular advantages and disadvantages of employing this methodological approach. The paper concludes with key pointers to aid those designing and appraising proposals for conducting case study research, and a checklist to help readers assess the quality of case study reports.

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Introduction

The case study approach is particularly useful to employ when there is a need to obtain an in-depth appreciation of an issue, event or phenomenon of interest, in its natural real-life context. Our aim in writing this piece is to provide insights into when to consider employing this approach and an overview of key methodological considerations in relation to the design, planning, analysis, interpretation and reporting of case studies.

The illustrative 'grand round', 'case report' and 'case series' have a long tradition in clinical practice and research. Presenting detailed critiques, typically of one or more patients, aims to provide insights into aspects of the clinical case and, in doing so, illustrate broader lessons that may be learnt. In research, the conceptually-related case study approach can be used, for example, to describe in detail a patient's episode of care, explore professional attitudes to and experiences of a new policy initiative or service development or more generally to 'investigate contemporary phenomena within its real-life context' [ 1 ]. Based on our experiences of conducting a range of case studies, we reflect on when to consider using this approach, discuss the key steps involved and illustrate, with examples, some of the practical challenges of attaining an in-depth understanding of a 'case' as an integrated whole. In keeping with previously published work, we acknowledge the importance of theory to underpin the design, selection, conduct and interpretation of case studies[ 2 ]. In so doing, we make passing reference to the different epistemological approaches used in case study research by key theoreticians and methodologists in this field of enquiry.

This paper is structured around the following main questions: What is a case study? What are case studies used for? How are case studies conducted? What are the potential pitfalls and how can these be avoided? We draw in particular on four of our own recently published examples of case studies (see Tables 1 , 2 , 3 and 4 ) and those of others to illustrate our discussion[ 3 – 7 ].

What is a case study?

A case study is a research approach that is used to generate an in-depth, multi-faceted understanding of a complex issue in its real-life context. It is an established research design that is used extensively in a wide variety of disciplines, particularly in the social sciences. A case study can be defined in a variety of ways (Table 5 ), the central tenet being the need to explore an event or phenomenon in depth and in its natural context. It is for this reason sometimes referred to as a "naturalistic" design; this is in contrast to an "experimental" design (such as a randomised controlled trial) in which the investigator seeks to exert control over and manipulate the variable(s) of interest.

Stake's work has been particularly influential in defining the case study approach to scientific enquiry. He has helpfully characterised three main types of case study: intrinsic , instrumental and collective [ 8 ]. An intrinsic case study is typically undertaken to learn about a unique phenomenon. The researcher should define the uniqueness of the phenomenon, which distinguishes it from all others. In contrast, the instrumental case study uses a particular case (some of which may be better than others) to gain a broader appreciation of an issue or phenomenon. The collective case study involves studying multiple cases simultaneously or sequentially in an attempt to generate a still broader appreciation of a particular issue.

These are however not necessarily mutually exclusive categories. In the first of our examples (Table 1 ), we undertook an intrinsic case study to investigate the issue of recruitment of minority ethnic people into the specific context of asthma research studies, but it developed into a instrumental case study through seeking to understand the issue of recruitment of these marginalised populations more generally, generating a number of the findings that are potentially transferable to other disease contexts[ 3 ]. In contrast, the other three examples (see Tables 2 , 3 and 4 ) employed collective case study designs to study the introduction of workforce reconfiguration in primary care, the implementation of electronic health records into hospitals, and to understand the ways in which healthcare students learn about patient safety considerations[ 4 – 6 ]. Although our study focusing on the introduction of General Practitioners with Specialist Interests (Table 2 ) was explicitly collective in design (four contrasting primary care organisations were studied), is was also instrumental in that this particular professional group was studied as an exemplar of the more general phenomenon of workforce redesign[ 4 ].

What are case studies used for?

According to Yin, case studies can be used to explain, describe or explore events or phenomena in the everyday contexts in which they occur[ 1 ]. These can, for example, help to understand and explain causal links and pathways resulting from a new policy initiative or service development (see Tables 2 and 3 , for example)[ 1 ]. In contrast to experimental designs, which seek to test a specific hypothesis through deliberately manipulating the environment (like, for example, in a randomised controlled trial giving a new drug to randomly selected individuals and then comparing outcomes with controls),[ 9 ] the case study approach lends itself well to capturing information on more explanatory ' how ', 'what' and ' why ' questions, such as ' how is the intervention being implemented and received on the ground?'. The case study approach can offer additional insights into what gaps exist in its delivery or why one implementation strategy might be chosen over another. This in turn can help develop or refine theory, as shown in our study of the teaching of patient safety in undergraduate curricula (Table 4 )[ 6 , 10 ]. Key questions to consider when selecting the most appropriate study design are whether it is desirable or indeed possible to undertake a formal experimental investigation in which individuals and/or organisations are allocated to an intervention or control arm? Or whether the wish is to obtain a more naturalistic understanding of an issue? The former is ideally studied using a controlled experimental design, whereas the latter is more appropriately studied using a case study design.

Case studies may be approached in different ways depending on the epistemological standpoint of the researcher, that is, whether they take a critical (questioning one's own and others' assumptions), interpretivist (trying to understand individual and shared social meanings) or positivist approach (orientating towards the criteria of natural sciences, such as focusing on generalisability considerations) (Table 6 ). Whilst such a schema can be conceptually helpful, it may be appropriate to draw on more than one approach in any case study, particularly in the context of conducting health services research. Doolin has, for example, noted that in the context of undertaking interpretative case studies, researchers can usefully draw on a critical, reflective perspective which seeks to take into account the wider social and political environment that has shaped the case[ 11 ].

How are case studies conducted?

Here, we focus on the main stages of research activity when planning and undertaking a case study; the crucial stages are: defining the case; selecting the case(s); collecting and analysing the data; interpreting data; and reporting the findings.

Defining the case

Carefully formulated research question(s), informed by the existing literature and a prior appreciation of the theoretical issues and setting(s), are all important in appropriately and succinctly defining the case[ 8 , 12 ]. Crucially, each case should have a pre-defined boundary which clarifies the nature and time period covered by the case study (i.e. its scope, beginning and end), the relevant social group, organisation or geographical area of interest to the investigator, the types of evidence to be collected, and the priorities for data collection and analysis (see Table 7 )[ 1 ]. A theory driven approach to defining the case may help generate knowledge that is potentially transferable to a range of clinical contexts and behaviours; using theory is also likely to result in a more informed appreciation of, for example, how and why interventions have succeeded or failed[ 13 ].

For example, in our evaluation of the introduction of electronic health records in English hospitals (Table 3 ), we defined our cases as the NHS Trusts that were receiving the new technology[ 5 ]. Our focus was on how the technology was being implemented. However, if the primary research interest had been on the social and organisational dimensions of implementation, we might have defined our case differently as a grouping of healthcare professionals (e.g. doctors and/or nurses). The precise beginning and end of the case may however prove difficult to define. Pursuing this same example, when does the process of implementation and adoption of an electronic health record system really begin or end? Such judgements will inevitably be influenced by a range of factors, including the research question, theory of interest, the scope and richness of the gathered data and the resources available to the research team.

Selecting the case(s)

The decision on how to select the case(s) to study is a very important one that merits some reflection. In an intrinsic case study, the case is selected on its own merits[ 8 ]. The case is selected not because it is representative of other cases, but because of its uniqueness, which is of genuine interest to the researchers. This was, for example, the case in our study of the recruitment of minority ethnic participants into asthma research (Table 1 ) as our earlier work had demonstrated the marginalisation of minority ethnic people with asthma, despite evidence of disproportionate asthma morbidity[ 14 , 15 ]. In another example of an intrinsic case study, Hellstrom et al.[ 16 ] studied an elderly married couple living with dementia to explore how dementia had impacted on their understanding of home, their everyday life and their relationships.

For an instrumental case study, selecting a "typical" case can work well[ 8 ]. In contrast to the intrinsic case study, the particular case which is chosen is of less importance than selecting a case that allows the researcher to investigate an issue or phenomenon. For example, in order to gain an understanding of doctors' responses to health policy initiatives, Som undertook an instrumental case study interviewing clinicians who had a range of responsibilities for clinical governance in one NHS acute hospital trust[ 17 ]. Sampling a "deviant" or "atypical" case may however prove even more informative, potentially enabling the researcher to identify causal processes, generate hypotheses and develop theory.

In collective or multiple case studies, a number of cases are carefully selected. This offers the advantage of allowing comparisons to be made across several cases and/or replication. Choosing a "typical" case may enable the findings to be generalised to theory (i.e. analytical generalisation) or to test theory by replicating the findings in a second or even a third case (i.e. replication logic)[ 1 ]. Yin suggests two or three literal replications (i.e. predicting similar results) if the theory is straightforward and five or more if the theory is more subtle. However, critics might argue that selecting 'cases' in this way is insufficiently reflexive and ill-suited to the complexities of contemporary healthcare organisations.

The selected case study site(s) should allow the research team access to the group of individuals, the organisation, the processes or whatever else constitutes the chosen unit of analysis for the study. Access is therefore a central consideration; the researcher needs to come to know the case study site(s) well and to work cooperatively with them. Selected cases need to be not only interesting but also hospitable to the inquiry [ 8 ] if they are to be informative and answer the research question(s). Case study sites may also be pre-selected for the researcher, with decisions being influenced by key stakeholders. For example, our selection of case study sites in the evaluation of the implementation and adoption of electronic health record systems (see Table 3 ) was heavily influenced by NHS Connecting for Health, the government agency that was responsible for overseeing the National Programme for Information Technology (NPfIT)[ 5 ]. This prominent stakeholder had already selected the NHS sites (through a competitive bidding process) to be early adopters of the electronic health record systems and had negotiated contracts that detailed the deployment timelines.

It is also important to consider in advance the likely burden and risks associated with participation for those who (or the site(s) which) comprise the case study. Of particular importance is the obligation for the researcher to think through the ethical implications of the study (e.g. the risk of inadvertently breaching anonymity or confidentiality) and to ensure that potential participants/participating sites are provided with sufficient information to make an informed choice about joining the study. The outcome of providing this information might be that the emotive burden associated with participation, or the organisational disruption associated with supporting the fieldwork, is considered so high that the individuals or sites decide against participation.

In our example of evaluating implementations of electronic health record systems, given the restricted number of early adopter sites available to us, we sought purposively to select a diverse range of implementation cases among those that were available[ 5 ]. We chose a mixture of teaching, non-teaching and Foundation Trust hospitals, and examples of each of the three electronic health record systems procured centrally by the NPfIT. At one recruited site, it quickly became apparent that access was problematic because of competing demands on that organisation. Recognising the importance of full access and co-operative working for generating rich data, the research team decided not to pursue work at that site and instead to focus on other recruited sites.

Collecting the data

In order to develop a thorough understanding of the case, the case study approach usually involves the collection of multiple sources of evidence, using a range of quantitative (e.g. questionnaires, audits and analysis of routinely collected healthcare data) and more commonly qualitative techniques (e.g. interviews, focus groups and observations). The use of multiple sources of data (data triangulation) has been advocated as a way of increasing the internal validity of a study (i.e. the extent to which the method is appropriate to answer the research question)[ 8 , 18 – 21 ]. An underlying assumption is that data collected in different ways should lead to similar conclusions, and approaching the same issue from different angles can help develop a holistic picture of the phenomenon (Table 2 )[ 4 ].

Brazier and colleagues used a mixed-methods case study approach to investigate the impact of a cancer care programme[ 22 ]. Here, quantitative measures were collected with questionnaires before, and five months after, the start of the intervention which did not yield any statistically significant results. Qualitative interviews with patients however helped provide an insight into potentially beneficial process-related aspects of the programme, such as greater, perceived patient involvement in care. The authors reported how this case study approach provided a number of contextual factors likely to influence the effectiveness of the intervention and which were not likely to have been obtained from quantitative methods alone.

In collective or multiple case studies, data collection needs to be flexible enough to allow a detailed description of each individual case to be developed (e.g. the nature of different cancer care programmes), before considering the emerging similarities and differences in cross-case comparisons (e.g. to explore why one programme is more effective than another). It is important that data sources from different cases are, where possible, broadly comparable for this purpose even though they may vary in nature and depth.

Analysing, interpreting and reporting case studies

Making sense and offering a coherent interpretation of the typically disparate sources of data (whether qualitative alone or together with quantitative) is far from straightforward. Repeated reviewing and sorting of the voluminous and detail-rich data are integral to the process of analysis. In collective case studies, it is helpful to analyse data relating to the individual component cases first, before making comparisons across cases. Attention needs to be paid to variations within each case and, where relevant, the relationship between different causes, effects and outcomes[ 23 ]. Data will need to be organised and coded to allow the key issues, both derived from the literature and emerging from the dataset, to be easily retrieved at a later stage. An initial coding frame can help capture these issues and can be applied systematically to the whole dataset with the aid of a qualitative data analysis software package.

The Framework approach is a practical approach, comprising of five stages (familiarisation; identifying a thematic framework; indexing; charting; mapping and interpretation) , to managing and analysing large datasets particularly if time is limited, as was the case in our study of recruitment of South Asians into asthma research (Table 1 )[ 3 , 24 ]. Theoretical frameworks may also play an important role in integrating different sources of data and examining emerging themes. For example, we drew on a socio-technical framework to help explain the connections between different elements - technology; people; and the organisational settings within which they worked - in our study of the introduction of electronic health record systems (Table 3 )[ 5 ]. Our study of patient safety in undergraduate curricula drew on an evaluation-based approach to design and analysis, which emphasised the importance of the academic, organisational and practice contexts through which students learn (Table 4 )[ 6 ].

Case study findings can have implications both for theory development and theory testing. They may establish, strengthen or weaken historical explanations of a case and, in certain circumstances, allow theoretical (as opposed to statistical) generalisation beyond the particular cases studied[ 12 ]. These theoretical lenses should not, however, constitute a strait-jacket and the cases should not be "forced to fit" the particular theoretical framework that is being employed.

When reporting findings, it is important to provide the reader with enough contextual information to understand the processes that were followed and how the conclusions were reached. In a collective case study, researchers may choose to present the findings from individual cases separately before amalgamating across cases. Care must be taken to ensure the anonymity of both case sites and individual participants (if agreed in advance) by allocating appropriate codes or withholding descriptors. In the example given in Table 3 , we decided against providing detailed information on the NHS sites and individual participants in order to avoid the risk of inadvertent disclosure of identities[ 5 , 25 ].

What are the potential pitfalls and how can these be avoided?

The case study approach is, as with all research, not without its limitations. When investigating the formal and informal ways undergraduate students learn about patient safety (Table 4 ), for example, we rapidly accumulated a large quantity of data. The volume of data, together with the time restrictions in place, impacted on the depth of analysis that was possible within the available resources. This highlights a more general point of the importance of avoiding the temptation to collect as much data as possible; adequate time also needs to be set aside for data analysis and interpretation of what are often highly complex datasets.

Case study research has sometimes been criticised for lacking scientific rigour and providing little basis for generalisation (i.e. producing findings that may be transferable to other settings)[ 1 ]. There are several ways to address these concerns, including: the use of theoretical sampling (i.e. drawing on a particular conceptual framework); respondent validation (i.e. participants checking emerging findings and the researcher's interpretation, and providing an opinion as to whether they feel these are accurate); and transparency throughout the research process (see Table 8 )[ 8 , 18 – 21 , 23 , 26 ]. Transparency can be achieved by describing in detail the steps involved in case selection, data collection, the reasons for the particular methods chosen, and the researcher's background and level of involvement (i.e. being explicit about how the researcher has influenced data collection and interpretation). Seeking potential, alternative explanations, and being explicit about how interpretations and conclusions were reached, help readers to judge the trustworthiness of the case study report. Stake provides a critique checklist for a case study report (Table 9 )[ 8 ].

Conclusions

The case study approach allows, amongst other things, critical events, interventions, policy developments and programme-based service reforms to be studied in detail in a real-life context. It should therefore be considered when an experimental design is either inappropriate to answer the research questions posed or impossible to undertake. Considering the frequency with which implementations of innovations are now taking place in healthcare settings and how well the case study approach lends itself to in-depth, complex health service research, we believe this approach should be more widely considered by researchers. Though inherently challenging, the research case study can, if carefully conceptualised and thoughtfully undertaken and reported, yield powerful insights into many important aspects of health and healthcare delivery.

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Acknowledgements

We are grateful to the participants and colleagues who contributed to the individual case studies that we have drawn on. This work received no direct funding, but it has been informed by projects funded by Asthma UK, the NHS Service Delivery Organisation, NHS Connecting for Health Evaluation Programme, and Patient Safety Research Portfolio. We would also like to thank the expert reviewers for their insightful and constructive feedback. Our thanks are also due to Dr. Allison Worth who commented on an earlier draft of this manuscript.

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AS conceived this article. SC, KC and AR wrote this paper with GH, AA and AS all commenting on various drafts. SC and AS are guarantors.

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Case study research for better evaluations of complex interventions: rationale and challenges

• Sara Paparini   ORCID: orcid.org/0000-0002-1909-2481 1 ,
• Judith Green 2 ,
• Chrysanthi Papoutsi 1 ,
• Jamie Murdoch 3 ,
• Mark Petticrew 4 ,
• Trish Greenhalgh 1 ,
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The need for better methods for evaluation in health research has been widely recognised. The ‘complexity turn’ has drawn attention to the limitations of relying on causal inference from randomised controlled trials alone for understanding whether, and under which conditions, interventions in complex systems improve health services or the public health, and what mechanisms might link interventions and outcomes. We argue that case study research—currently denigrated as poor evidence—is an under-utilised resource for not only providing evidence about context and transferability, but also for helping strengthen causal inferences when pathways between intervention and effects are likely to be non-linear.

Case study research, as an overall approach, is based on in-depth explorations of complex phenomena in their natural, or real-life, settings. Empirical case studies typically enable dynamic understanding of complex challenges and provide evidence about causal mechanisms and the necessary and sufficient conditions (contexts) for intervention implementation and effects. This is essential evidence not just for researchers concerned about internal and external validity, but also research users in policy and practice who need to know what the likely effects of complex programmes or interventions will be in their settings. The health sciences have much to learn from scholarship on case study methodology in the social sciences. However, there are multiple challenges in fully exploiting the potential learning from case study research. First are misconceptions that case study research can only provide exploratory or descriptive evidence. Second, there is little consensus about what a case study is, and considerable diversity in how empirical case studies are conducted and reported. Finally, as case study researchers typically (and appropriately) focus on thick description (that captures contextual detail), it can be challenging to identify the key messages related to intervention evaluation from case study reports.

Whilst the diversity of published case studies in health services and public health research is rich and productive, we recommend further clarity and specific methodological guidance for those reporting case study research for evaluation audiences.

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The need for methodological development to address the most urgent challenges in health research has been well-documented. Many of the most pressing questions for public health research, where the focus is on system-level determinants [ 1 , 2 ], and for health services research, where provisions typically vary across sites and are provided through interlocking networks of services [ 3 ], require methodological approaches that can attend to complexity. The need for methodological advance has arisen, in part, as a result of the diminishing returns from randomised controlled trials (RCTs) where they have been used to answer questions about the effects of interventions in complex systems [ 4 , 5 , 6 ]. In conditions of complexity, there is limited value in maintaining the current orientation to experimental trial designs in the health sciences as providing ‘gold standard’ evidence of effect.

There are increasing calls for methodological pluralism [ 7 , 8 ], with the recognition that complex intervention and context are not easily or usefully separated (as is often the situation when using trial design), and that system interruptions may have effects that are not reducible to linear causal pathways between intervention and outcome. These calls are reflected in a shifting and contested discourse of trial design, seen with the emergence of realist [ 9 ], adaptive and hybrid (types 1, 2 and 3) [ 10 , 11 ] trials that blend studies of effectiveness with a close consideration of the contexts of implementation. Similarly, process evaluation has now become a core component of complex healthcare intervention trials, reflected in MRC guidance on how to explore implementation, causal mechanisms and context [ 12 ].

Evidence about the context of an intervention is crucial for questions of external validity. As Woolcock [ 4 ] notes, even if RCT designs are accepted as robust for maximising internal validity, questions of transferability (how well the intervention works in different contexts) and generalisability (how well the intervention can be scaled up) remain unanswered [ 5 , 13 ]. For research evidence to have impact on policy and systems organisation, and thus to improve population and patient health, there is an urgent need for better methods for strengthening external validity, including a better understanding of the relationship between intervention and context [ 14 ].

Policymakers, healthcare commissioners and other research users require credible evidence of relevance to their settings and populations [ 15 ], to perform what Rosengarten and Savransky [ 16 ] call ‘careful abstraction’ to the locales that matter for them. They also require robust evidence for understanding complex causal pathways. Case study research, currently under-utilised in public health and health services evaluation, can offer considerable potential for strengthening faith in both external and internal validity. For example, in an empirical case study of how the policy of free bus travel had specific health effects in London, UK, a quasi-experimental evaluation (led by JG) identified how important aspects of context (a good public transport system) and intervention (that it was universal) were necessary conditions for the observed effects, thus providing useful, actionable evidence for decision-makers in other contexts [ 17 ].

The overall approach of case study research is based on the in-depth exploration of complex phenomena in their natural, or ‘real-life’, settings. Empirical case studies typically enable dynamic understanding of complex challenges rather than restricting the focus on narrow problem delineations and simple fixes. Case study research is a diverse and somewhat contested field, with multiple definitions and perspectives grounded in different ways of viewing the world, and involving different combinations of methods. In this paper, we raise awareness of such plurality and highlight the contribution that case study research can make to the evaluation of complex system-level interventions. We review some of the challenges in exploiting the current evidence base from empirical case studies and conclude by recommending that further guidance and minimum reporting criteria for evaluation using case studies, appropriate for audiences in the health sciences, can enhance the take-up of evidence from case study research.

Case study research offers evidence about context, causal inference in complex systems and implementation

Well-conducted and described empirical case studies provide evidence on context, complexity and mechanisms for understanding how, where and why interventions have their observed effects. Recognition of the importance of context for understanding the relationships between interventions and outcomes is hardly new. In 1943, Canguilhem berated an over-reliance on experimental designs for determining universal physiological laws: ‘As if one could determine a phenomenon’s essence apart from its conditions! As if conditions were a mask or frame which changed neither the face nor the picture!’ ([ 18 ] p126). More recently, a concern with context has been expressed in health systems and public health research as part of what has been called the ‘complexity turn’ [ 1 ]: a recognition that many of the most enduring challenges for developing an evidence base require a consideration of system-level effects [ 1 ] and the conceptualisation of interventions as interruptions in systems [ 19 ].

The case study approach is widely recognised as offering an invaluable resource for understanding the dynamic and evolving influence of context on complex, system-level interventions [ 20 , 21 , 22 , 23 ]. Empirically, case studies can directly inform assessments of where, when, how and for whom interventions might be successfully implemented, by helping to specify the necessary and sufficient conditions under which interventions might have effects and to consolidate learning on how interdependencies, emergence and unpredictability can be managed to achieve and sustain desired effects. Case study research has the potential to address four objectives for improving research and reporting of context recently set out by guidance on taking account of context in population health research [ 24 ], that is to (1) improve the appropriateness of intervention development for specific contexts, (2) improve understanding of ‘how’ interventions work, (3) better understand how and why impacts vary across contexts and (4) ensure reports of intervention studies are most useful for decision-makers and researchers.

However, evaluations of complex healthcare interventions have arguably not exploited the full potential of case study research and can learn much from other disciplines. For evaluative research, exploratory case studies have had a traditional role of providing data on ‘process’, or initial ‘hypothesis-generating’ scoping, but might also have an increasing salience for explanatory aims. Across the social and political sciences, different kinds of case studies are undertaken to meet diverse aims (description, exploration or explanation) and across different scales (from small N qualitative studies that aim to elucidate processes, or provide thick description, to more systematic techniques designed for medium-to-large N cases).

Case studies with explanatory aims vary in terms of their positioning within mixed-methods projects, with designs including (but not restricted to) (1) single N of 1 studies of interventions in specific contexts, where the overall design is a case study that may incorporate one or more (randomised or not) comparisons over time and between variables within the case; (2) a series of cases conducted or synthesised to provide explanation from variations between cases; and (3) case studies of particular settings within RCT or quasi-experimental designs to explore variation in effects or implementation.

Detailed qualitative research (typically done as ‘case studies’ within process evaluations) provides evidence for the plausibility of mechanisms [ 25 ], offering theoretical generalisations for how interventions may function under different conditions. Although RCT designs reduce many threats to internal validity, the mechanisms of effect remain opaque, particularly when the causal pathways between ‘intervention’ and ‘effect’ are long and potentially non-linear: case study research has a more fundamental role here, in providing detailed observational evidence for causal claims [ 26 ] as well as producing a rich, nuanced picture of tensions and multiple perspectives [ 8 ].

Longitudinal or cross-case analysis may be best suited for evidence generation in system-level evaluative research. Turner [ 27 ], for instance, reflecting on the complex processes in major system change, has argued for the need for methods that integrate learning across cases, to develop theoretical knowledge that would enable inferences beyond the single case, and to develop generalisable theory about organisational and structural change in health systems. Qualitative Comparative Analysis (QCA) [ 28 ] is one such formal method for deriving causal claims, using set theory mathematics to integrate data from empirical case studies to answer questions about the configurations of causal pathways linking conditions to outcomes [ 29 , 30 ].

Nonetheless, the single N case study, too, provides opportunities for theoretical development [ 31 ], and theoretical generalisation or analytical refinement [ 32 ]. How ‘the case’ and ‘context’ are conceptualised is crucial here. Findings from the single case may seem to be confined to its intrinsic particularities in a specific and distinct context [ 33 ]. However, if such context is viewed as exemplifying wider social and political forces, the single case can be ‘telling’, rather than ‘typical’, and offer insight into a wider issue [ 34 ]. Internal comparisons within the case can offer rich possibilities for logical inferences about causation [ 17 ]. Further, case studies of any size can be used for theory testing through refutation [ 22 ]. The potential lies, then, in utilising the strengths and plurality of case study to support theory-driven research within different methodological paradigms.

Evaluation research in health has much to learn from a range of social sciences where case study methodology has been used to develop various kinds of causal inference. For instance, Gerring [ 35 ] expands on the within-case variations utilised to make causal claims. For Gerring [ 35 ], case studies come into their own with regard to invariant or strong causal claims (such as X is a necessary and/or sufficient condition for Y) rather than for probabilistic causal claims. For the latter (where experimental methods might have an advantage in estimating effect sizes), case studies offer evidence on mechanisms: from observations of X affecting Y, from process tracing or from pattern matching. Case studies also support the study of emergent causation, that is, the multiple interacting properties that account for particular and unexpected outcomes in complex systems, such as in healthcare [ 8 ].

Finally, efficacy (or beliefs about efficacy) is not the only contributor to intervention uptake, with a range of organisational and policy contingencies affecting whether an intervention is likely to be rolled out in practice. Case study research is, therefore, invaluable for learning about contextual contingencies and identifying the conditions necessary for interventions to become normalised (i.e. implemented routinely) in practice [ 36 ].

The challenges in exploiting evidence from case study research

At present, there are significant challenges in exploiting the benefits of case study research in evaluative health research, which relate to status, definition and reporting. Case study research has been marginalised at the bottom of an evidence hierarchy, seen to offer little by way of explanatory power, if nonetheless useful for adding descriptive data on process or providing useful illustrations for policymakers [ 37 ]. This is an opportune moment to revisit this low status. As health researchers are increasingly charged with evaluating ‘natural experiments’—the use of face masks in the response to the COVID-19 pandemic being a recent example [ 38 ]—rather than interventions that take place in settings that can be controlled, research approaches using methods to strengthen causal inference that does not require randomisation become more relevant.

A second challenge for improving the use of case study evidence in evaluative health research is that, as we have seen, what is meant by ‘case study’ varies widely, not only across but also within disciplines. There is indeed little consensus amongst methodologists as to how to define ‘a case study’. Definitions focus, variously, on small sample size or lack of control over the intervention (e.g. [ 39 ] p194), on in-depth study and context [ 40 , 41 ], on the logic of inference used [ 35 ] or on distinct research strategies which incorporate a number of methods to address questions of ‘how’ and ‘why’ [ 42 ]. Moreover, definitions developed for specific disciplines do not capture the range of ways in which case study research is carried out across disciplines. Multiple definitions of case study reflect the richness and diversity of the approach. However, evidence suggests that a lack of consensus across methodologists results in some of the limitations of published reports of empirical case studies [ 43 , 44 ]. Hyett and colleagues [ 43 ], for instance, reviewing reports in qualitative journals, found little match between methodological definitions of case study research and how authors used the term.

This raises the third challenge we identify that case study reports are typically not written in ways that are accessible or useful for the evaluation research community and policymakers. Case studies may not appear in journals widely read by those in the health sciences, either because space constraints preclude the reporting of rich, thick descriptions, or because of the reported lack of willingness of some biomedical journals to publish research that uses qualitative methods [ 45 ], signalling the persistence of the aforementioned evidence hierarchy. Where they do, however, the term ‘case study’ is used to indicate, interchangeably, a qualitative study, an N of 1 sample, or a multi-method, in-depth analysis of one example from a population of phenomena. Definitions of what constitutes the ‘case’ are frequently lacking and appear to be used as a synonym for the settings in which the research is conducted. Despite offering insights for evaluation, the primary aims may not have been evaluative, so the implications may not be explicitly drawn out. Indeed, some case study reports might properly be aiming for thick description without necessarily seeking to inform about context or causality.

Acknowledging plurality and developing guidance

We recognise that definitional and methodological plurality is not only inevitable, but also a necessary and creative reflection of the very different epistemological and disciplinary origins of health researchers, and the aims they have in doing and reporting case study research. Indeed, to provide some clarity, Thomas [ 46 ] has suggested a typology of subject/purpose/approach/process for classifying aims (e.g. evaluative or exploratory), sample rationale and selection and methods for data generation of case studies. We also recognise that the diversity of methods used in case study research, and the necessary focus on narrative reporting, does not lend itself to straightforward development of formal quality or reporting criteria.

Existing checklists for reporting case study research from the social sciences—for example Lincoln and Guba’s [ 47 ] and Stake’s [ 33 ]—are primarily orientated to the quality of narrative produced, and the extent to which they encapsulate thick description, rather than the more pragmatic issues of implications for intervention effects. Those designed for clinical settings, such as the CARE (CAse REports) guidelines, provide specific reporting guidelines for medical case reports about single, or small groups of patients [ 48 ], not for case study research.

The Design of Case Study Research in Health Care (DESCARTE) model [ 44 ] suggests a series of questions to be asked of a case study researcher (including clarity about the philosophy underpinning their research), study design (with a focus on case definition) and analysis (to improve process). The model resembles toolkits for enhancing the quality and robustness of qualitative and mixed-methods research reporting, and it is usefully open-ended and non-prescriptive. However, even if it does include some reflections on context, the model does not fully address aspects of context, logic and causal inference that are perhaps most relevant for evaluative research in health.

Hence, for evaluative research where the aim is to report empirical findings in ways that are intended to be pragmatically useful for health policy and practice, this may be an opportune time to consider how to best navigate plurality around what is (minimally) important to report when publishing empirical case studies, especially with regards to the complex relationships between context and interventions, information that case study research is well placed to provide.

The conventional scientific quest for certainty, predictability and linear causality (maximised in RCT designs) has to be augmented by the study of uncertainty, unpredictability and emergent causality [ 8 ] in complex systems. This will require methodological pluralism, and openness to broadening the evidence base to better understand both causality in and the transferability of system change intervention [ 14 , 20 , 23 , 25 ]. Case study research evidence is essential, yet is currently under exploited in the health sciences. If evaluative health research is to move beyond the current impasse on methods for understanding interventions as interruptions in complex systems, we need to consider in more detail how researchers can conduct and report empirical case studies which do aim to elucidate the contextual factors which interact with interventions to produce particular effects. To this end, supported by the UK’s Medical Research Council, we are embracing the challenge to develop guidance for case study researchers studying complex interventions. Following a meta-narrative review of the literature, we are planning a Delphi study to inform guidance that will, at minimum, cover the value of case study research for evaluating the interrelationship between context and complex system-level interventions; for situating and defining ‘the case’, and generalising from case studies; as well as provide specific guidance on conducting, analysing and reporting case study research. Our hope is that such guidance can support researchers evaluating interventions in complex systems to better exploit the diversity and richness of case study research.

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Abbreviations

Qualitative comparative analysis

Quasi-experimental design

Randomised controlled trial

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This work was funded by the Medical Research Council - MRC Award MR/S014632/1 HCS: Case study, Context and Complex interventions (TRIPLE C). SP was additionally funded by the University of Oxford's Higher Education Innovation Fund (HEIF).

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Paparini, S., Green, J., Papoutsi, C. et al. Case study research for better evaluations of complex interventions: rationale and challenges. BMC Med 18 , 301 (2020). https://doi.org/10.1186/s12916-020-01777-6

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Using the Matrixed Multiple Case Study approach to identify factors affecting the uptake of IPV screening programs following the use of implementation facilitation

• Omonyêlé L. Adjognon   ORCID: orcid.org/0000-0002-0674-089X 1 ,
• Julianne E. Brady 1 ,
• Katherine M. Iverson 2 , 3 ,
• Kelly Stolzmann 1 ,
• Melissa E. Dichter 4 , 5 ,
• Robert A. Lew 6 ,
• Megan R. Gerber 7 , 8 ,
• Galina A. Portnoy 9 , 10 ,
• Samina Iqbal 11 , 12 ,
• Sally G. Haskell 9 , 13 , 14 ,
• Le Ann E. Bruce 15 , 16 &
• Christopher J. Miller 1 , 17  

Implementation Science Communications volume  4 , Article number:  145 ( 2023 ) Cite this article

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Intimate partner violence (IPV) is a prevalent social determinant of health. The US Preventive Services Task Force recommends routine IPV screening of women, but uptake remains variable. The Veterans Health Administration (VHA) initiated implementation facilitation (IF) to support integration of IPV screening programs into primary care clinics. An evaluation of IF efforts showed variability in IPV screening rates across sites. The follow-up study presented here used a Matrixed Multiple Case Study (MMCS) approach to examine the multilevel factors impacting IPV screening program implementation across sites with varying levels of implementation success.

This mixed methods study is part of a larger cluster randomized stepped wedge Hybrid-II program evaluation. In the larger trial, participating sites received 6 months of IF consisting of an external facilitator from VHA’s Office of Women’s Health working closely with an internal facilitator and key site personnel. Recognizing the heterogeneity in implementation outcomes across sites, the MMCS approach was used to enable interpretation of qualitative and quantitative data within and across sites to help contextualize the primary findings from the larger study. Qualitative data collection was guided by the integrated Promoting Action on Research Implementation in Health Services (i-PARIHS) framework and included interviews with key informants involved in IPV screening implementation at eight sites. Quantitative data on IPV screening uptake was derived from medical records and surveys completed by key personnel at the same eight sites to understand implementation facilitation activities.

Fifteen factors influencing IPV screening implementation spanning all four i-PARIHS domains were identified and categorized into three distinct categories: (1) factors with enabling influence across all sites, (2) factors deemed important to implementation success, and (3) factors differentiating sites with high/medium versus low implementation success.

Conclusions

Understanding the influencing factors across multi-level domains contributing to variable success of IPV screening implementation can inform the tailoring of IF efforts to promote spread and quality of screening. Implementation of IPV screening programs in primary care with IF should consider consistent engagement of internal facilitators with clinic staff involved in implementation, the resourcefulness of external facilitators, and appending resources to IPV screening tools to help key personnel address positive screens.

Trial registration

ClinicalTrials.gov NCT04106193. Registered on September 26, 2019.

Peer Review reports

Contributions to the literature

A quantitative evaluation showed an overall increase in screening rates in primary care clinics for intimate partner violence (IPV) using implementation facilitation (IF) but did not fully explain across-site variation.

The Matrixed Multiple Case Study (MMCS) is a novel approach to identify site-specific factors that influence implementation success.

This is the first evaluation using MMCS to understand factors influencing IPV screening implementation and provide a set of factors to consider to maximize implementation success.

Applying lessons learned from these analyses can help provide consistent IPV screening, especially in primary care where women who experience IPV frequently receive care.

Intimate partner violence (IPV) is defined as physical or sexual violence, stalking, or psychological aggression by a past or current intimate partner [ 1 ]. Although IPV can affect persons of any gender, women are at an increased risk of experiencing IPV and associated physical [ 2 , 3 , 4 ], psychological [ 3 , 5 , 6 ], and social health issues [ 4 , 7 ]. With nearly 640 million women worldwide experiencing IPV during their lifetime [ 8 , 9 , 10 ], identifying and addressing IPV is an important public health issue.

The poorer health status of women experiencing IPV often results in increased healthcare use [ 11 , 12 ], specifically within the primary care setting where women experiencing IPV commonly present for care [ 11 , 13 ]. Healthcare settings—and primary care in particular—represent ideal places to identify women who may be experiencing IPV so appropriate resources can be offered [ 8 , 10 ]. The U.S. Preventive Services Task Force (USPSTF) recommends routine screening for women of childbearing age [ 14 , 15 ]. The Veterans Health Administration (VHA) [ 16 ] recommends IPV screening annually for all Veterans regardless of gender or age, but, per policy (VHA Directive 1198), recognizes Veterans who identify as women are at a higher risk for IPV than their civilian counterparts [ 4 ] and requires (at a minimum) annual screening of all women of childbearing age consistent with the USPSTF recommendations. In addition, a national IPV screening protocol, which includes a standardized 5-item screening tool [ 17 , 18 ], has been disseminated to all VHA healthcare facilities as a template in the electronic health record.

Despite these recommendations, requirements, and protocols, screening uptake in clinical settings remains variable both inside and outside of VHA due to barriers including, but not limited to, lack of provider training, time constraints, and providers’ discomfort discussing IPV with patients [ 19 , 20 , 21 , 22 , 23 , 24 ]. To address these barriers and improve uptake of IPV screening and response practices, VHA’s Office of Women’s Health (OWH) initiated implementation facilitation (IF [ 25 , 26 ]) via a stepped wedge hybrid implementation-effectiveness trial at nine VHA clinical sites throughout the US [ 27 ].

Primary outcomes from the evaluation of these implementation efforts [ 28 ] in the larger clinical trial suggested that IF was associated with substantial increases in reach of screening at these sites, including nearly doubling the number of women identified as having experienced past-year IPV (as previously reported [ 28 ]). The increased identification of women experiencing IPV in turn enabled linkages with support services (e.g., social work or mental health).

These aggregate findings, however, do not account for substantial site-to-site variability in implementation success, nor point to what specific factors may have differentiated high- from low-implementation success sites within the trial. Understanding specific factors that influence implementation success using IF can help tailor the intervention for effective IPV screening implementation. In this follow-up study, we describe the application of a Matrixed Multiple Case Study (MMCS) approach [ 29 ] to analyze and interpret the primary findings from our trial [ 28 ], which included substantial variability in increasing reach of IPV screening programs, with respect to factors that influenced implementation success. This methodology allows for the examination of multiple aspects of the implementation process to understand the combination of factors that are associated with the successful implementation of evidence-based practices—in this case, IPV screening programs.

Details of the design and primary outcomes of the trial are reported elsewhere [ 27 , 28 ]. For this follow-up study, we provide brief descriptions of the data sources and the steps taken by the study team in the application of the MMCS approach used for analyses. The VA Boston Healthcare System Institutional Review Board approved this study.

Site recruitment

As part of the larger trial, nine sites from across the United States were recruited by VHA’s Office of Women’s Health. Local leadership at each site signed a project letter agreement for enrollment (see Iverson et al. (2023) [ 28 ] and Supplemental file 1 for more details). One of these sites was not included in this follow-up analysis due to exceptionally high IPV screening rates prior to the start of the implementation facilitation period.

Participants

This mixed-methods follow-up evaluation engaged various participants. These included VHA staff involved with IPV screening program implementation at each site (e.g., primary care providers, nurses, IPVAP Coordinator, or designee), as well as off-site external facilitators from OWH who worked with staff at each site to support the implementation of IPV screening programs. In addition, medical record data for all women receiving primary care services in the participating clinics at the enrolled sites 3 months prior to (i.e., pre-implementation period) and 9 months after implementation facilitation was initiated (i.e., implementation period) were included in this analysis.

Intervention

In the primary study, each site received 6 months of implementation facilitation (IF). IF involved trained external facilitators from OWH working closely with local internal facilitators to help integrate IPV screening programs at the participating sites [ 25 , 26 ]. Internal facilitators came from myriad clinical and training backgrounds, including primary care physicians, nurses, and IPVAP Coordinators. IPVAP Coordinators are responsible for providing training, education, and consultation to clinical staff on IPV screening, response, and referral practices as part of VHA’s standard implementation protocol. IF activities included multi-faceted, personalized support via regular phone and video conferencing meetings and ad-hoc asynchronous and synchronous communication via messaging and email. These activities aimed to provide education and address implementation challenges, with the common goal of integrating IPV screening and response programs during a 6-month period at each site.

Data collection to inform the MMCS approach

Table 1 describes the six distinct quantitative and qualitative data sources that informed the MMCS analyses. In short, implementation success for each site (i.e., the dependent variable) was determined by an aggregate of four variables derived from one data source, while data on potential influencing factors (i.e., the independent variables) were derived from an additional five data sources.

Data analysis

Following the completion of the larger clinical trial, we used the MMCS approach [ 29 ] presented here, which compares site-specific matrices containing information from qualitative and quantitative data sources across and within sites, enabling the emergence of generalizable knowledge from common and heterogenous local factors influencing the success of program implementation across sites. See Fig. 1 for a process map summarizing the nine steps of the MMCS as applied to this evaluation.

MMCS analytic process

MMCS analyses began with describing the research goal (MMCS Step 1) and used an aggregate of four site-level variables to define implementation success (MMCS Step 2). These included (a) change in reach of IPV screening efforts, along with (b) the consent rate for IPV screening at each site (i.e., percent of women offered screening, via an annual clinical reminder in the electronic health record, who consented to complete the screen). We included consent rate as an important component of the success of a screening program because, at some sites, an unrealistically high percentage of women who were offered screening were marked in the electronic medical record as either declining to complete the screening or not being able to complete the screening due to the presence of another family member (i.e., non-consent). Thus, sites with a high non-consent rate (e.g., above 50%) were deemed to have lower implementation success. Other variables defining implementation success included (c) IPV disclosure rate and (d) post-screening psychosocial service use. Very low disclosure rates (e.g., below 5% of completed screens) could indicate that screening was not conducted in a thoughtful or sensitive manner, while very low rates of psychosocial service use among women screening positive for IPV could indicate that these women were not offered follow-up services. For details on how the study team used these variables to assess the extent of implementation success per site, see MMCS Step 5.

To select relevant domains of factors influencing implementation (MMCS Step 3), the study team reviewed data from one site and developed a list of potentially important influencing factors within each domain of the i-PARIHS framework [ 30 ]. The i-PARIHS framework uses four domains (i.e., facilitation , innovation , recipients , context ) to explain complex implementation of research into clinical practice. The influencing factor list was refined during a series of consensus meetings and application to other participating sites. Once these data sources and variables were identified, the study team gathered the data for each domain (MMCS Step 4). The study team engaged in MMCS Steps 5 and 6 in parallel. For MMCS Step 5, they independently rated each site's implementation success status as high, medium, or low based on the four site-level variables. Disagreements in this process were then resolved via a consensus meeting where team members discussed discrepancies until they reached mutual agreement. This process resulted in the final implementation success ranking of sites. Due to the low number of sites included in the analysis, the study team chose to dichotomize the implementation success statuses as high/medium or low performing, resulting in all eight sites being placed into one of these two categories.

For MMCS Step 6, two study team members (OLA, JEB) used five data sources (from Table 1: implementation strategies survey, site balancing characteristics, time-motion tracker, stakeholder interviews, and external facilitator interviews) to summarize the influencing factors in each domain per site. This included determining (a) the factor’s relative presence at the site, and (b) the factor’s impact on implementation success at that site. These two team members were blinded to the site implementation success status for this part of the analytic process, and they met frequently to establish consensus. Next, the data were organized into site-specific sortable matrices (MMCS Step 7), and the team completed within-site analysis (MMCS Step 8). For this within-site analysis, the study team assessed the status and influence of factors on implementation success and then aggregated the factors and influences into a sortable cross-site matrix. Once the cross-site matrix was assembled, cross-site analysis was conducted to determine (a) factors universally present and enabling across all sites, (b) factors with a strong relationship between presence and enablement of implementation success, and (c) factors that differentiated sites by overall implementation success (MMCS Step 9). Discrepancies in categorizations among team members were resolved via a series of consensus meetings where these discrepancies were discussed until the team reached mutual agreement, resulting in the identification of the fifteen influencing factors impacting implementation success.

Participant characteristics

Medical record data for all women ( n = 5149) seen in the participating primary care clinics during the pre-implementation and implementation facilitation period were included in the ranking of sites’ implementation success. Table 2 presents screening rates showing site-by-site variability. The study team ranked four sites as having high/medium implementation success and four sites as having low implementation success.

Implementation strategies survey respondents (1–3 per site) represented all eight sites and included eight IPVAP Coordinators, four primary care providers, and three nurses. Semi-structured qualitative interviews were conducted post-IF with 14 internal facilitators and persons closely involved with implementation facilitation (1–2 per site) at the eight sites from the larger study [ 28 ]. Interviewees included eight IPVAP Coordinators, five primary care providers, and one nurse.

Types of influencing factors

We identified 15 factors affecting the success of IPV screening program implementation across sites. These factors span all i-PARIHS domains: four facilitation , two innovation , four recipients , and five context factors. As summarized in Table 3 , we present factors by influencing characteristics (i.e., presence and influence) in relationship to implementation success status and by the i-PARIHS domains.

Factors with enabling influence across all sites

Three factors were present across all sites regardless of implementation success status, that consistently enabled IPV screening program implementation. These factors are from the facilitation and recipients domains.

In the facilitation domain, these factors were internal facilitator or other site staff are available to meet and communicate regularly , and internal facilitator or IPVAP Coordinator organizes/conducts staff training for IPV screening . More specifically, all sites indicated that having key staff involved in the implementation process available and consistently engaged through both regular communication and training were foundational to successful implementation.

In the recipients domain, the influencing factor was IPVAP Coordinator is available and actively engaged in supporting IPV screening day-to-day implementation activities at main hospital and/or connected community-based outpatient clinics. The factor extends from the availability of key staff assisting with implementation, to focus on the IPVAP Coordinator’s daily actions engaging key medical center personnel in implementation.

Factors deemed important to implementation success

Six factors emerged such that their presence was enabling, and their absence hindering to implementation—but unlike the previous section, these factors were not present at all sites. Rather, when they were present, they enabled IPV screening implementation, but their absence hindered IPV screening implementation. These factors were identified in all four i-PARIHS domains.

In the facilitation domain, the two influencing factors are external facilitator is available and willing to meet and communicate regularly , and external facilitator is perceived as knowledgeable about IPV screening practices and available resources . The value of external facilitators to support standing up IPV screening programs is based on both the perceived expertise and resourcefulness of the external facilitator, and the external facilitators regular engagement with the site.

In the innovation domain, sites where IPV screening is seen as duplicative with other established clinical reminders faced more challenges to implementation. Conversely, sites were better able to implement IPV screening when this perceived duplication was absent.

In the recipients domain, three factors present a strong relationship between presence and enablement across sites regardless of implementation success status. These are: Primary care team is available to screen for IPV during patient visits ; frontline staff have the expertise and available resources to conduct and follow-up IPV screening as intended (e.g., trained primary care social workers) ; and at least one frontline primary care staff person is supportive of IPV screening in the identified clinics. The availability of primary care clinic members (e.g., nursing staff, primary care providers, social workers) that are educated in screening and response practices and passionate about implementing IPV screening programs was seen as important for site staff but were not sufficient by themselves to ensure implementation success.

In the context domain, we identified five influencing factors: Site has an engaged information technology (IT) team or infrastructure in place to support IPV screening prior to initiation of IF activities ; site-level p rimary care, women's health, social work, and/or medical center leadership is supportive of IPV screening in identified clinic(s) ; competing priorities created barriers to prioritizing IPV screening implementation (e.g., COVID-19, other initiatives) ; regional and/or national leadership is supportive of IPV screening , and access to community resources for additional support for patients. Multilevel leadership support outside of the primary care clinic was viewed as beneficial to moving the implementation process along and for increasing buy-in among clinic staff. Similarly, when staff felt that cross-service (e.g., nursing and social work) and multi-level leadership (e.g., service chiefs, medical center directors, regional leaders) was invested in the implementation process (e.g., by providing staff support and/or protected time for IPV screening activities) , increased buy-in was reported and staff were more resilient to overcoming barriers faced during the implementation process.

Factors differentiating sites with high/medium versus low implementation success

Only one influencing factor in the innovation domain differentiated the sites with high/medium from sites with low implementation success. This factor, IPV screening clinical reminder is designed to easily access appropriate follow-up services if someone screens positive, was strongly present and enabling in most high/medium implementation success sites but seldom in the low implementation success sites. This suggests that if the screening protocol did not include clear guidance and easy pathways for referral options and resources, sites were less comfortable implementing IPV screening in the clinic, thereby leading to lower overall implementation success.

When implemented successfully, IPV screening programs are effective in identifying women who experience IPV for provision of resources and support services [ 4 ]. Primary study outcomes showed that using implementation facilitation as a strategy to scale up IPV screening implementation resulted in increased IPV screening rates and identification of patients experiencing IPV [ 28 ]. Nonetheless, aggregate IPV screening implementation outcomes on their own cannot explain site-level variability in screening rates, and therefore portray an incomplete account of the factors that may ultimately be responsible for implementation success at some sites—but shortfalls at other sites. The use of the MMCS approach [ 29 ] and the i-PARIHS framework for this follow-up study enabled a deeper analysis to identify factors that contribute to the success of IPV screening implementation among primary care clinics that participated in the clinical trial [ 28 ]. This study identified 15 factors that influence the success of IPV screening program implementation in these primary care clinics.

Overall, no single influencing factor carries enough weight to guarantee IPV screening implementation success. Influencing factors presented here should be carefully considered in tandem to overcome the known barriers to IPV screening implementation (e.g., time constraints, lack of clinician training, and discomfort addressing IPV) [ 19 , 20 , 21 , 22 , 23 , 24 ]. These factors and their respective associations with implementation success provide insights across all domains of the i-PARIHS framework. Influencing factors in the innovation domain suggest the importance of establishing two key components prior to IPV screening program implementation: clearly and effectively communicating to all primary care clinic staff the importance of integrating IPV screening programs into routine care, and delineating the distinct nature of IPV screening from other existing screens (e.g., broader interpersonal violence screening) to avoid perceptions of duplication with other screening efforts used in the clinic. Presumably, these can potentially be achieved through a variety of methods including clinician education [ 31 ], audit and feedback [ 32 ], or pay-for-performance incentives [ 33 ] that demonstrate the health system’s commitment to this type of screening. In addition, ensuring that the IPV screening protocol contains clear guidance on effectively responding to positive screens, particularly in terms of accessible resources and easily being able to refer patients with support services, is crucial. Ideally, this would be integrated in the screening protocol template embedded within the electronic medical records so that options for referral are immediately available following positive screens [ 34 , 35 ], as clinicians’ lack of knowledge or the availability of referral options and resources has previously emerged as a significant barrier to routine IPV screening [ 23 , 24 , 34 , 36 , 37 ]. In our analysis, the range in availability and quality of referral options and resources across sites suggested that sites with more robust referral pathways and resources were better equipped for implementation success than sites with minimal resources readily available. Universally building these robust resources into the IPV screening tool itself could help equip staff and providers to respond adequately to positive screens, thereby bolstering confidence and encouraging buy-in, which the literature shows is a key facilitator of IPV screening and response practices [ 34 , 37 , 38 ].

When we closely examine findings within the context and recipients domains, our findings speak to the importance of establishing foundational enabling factors to increase the likelihood of successful implementation of IPV screening programs. First, our findings suggest that it is important to identify key implementation staff (an internal facilitator and other clinic staff) who are able and willing to engage consistently through regular communication and training. Second, getting cross-service and multi-level leadership buy-in into the implementation process itself (e.g., giving staff protected time to dedicate to implementation activities) provides the implementation teams the necessary support to address and overcome implementation barriers. These findings replicate and extend past studies [ 22 , 24 , 39 , 40 ].

With facilitation, influencing factors suggest that IF is helpful to IPV screening implementation when it is led by resourceful external facilitators with high levels of knowledge and experience with IPV screening, who thoughtfully and regularly engage with an internal facilitator and other members of the implementation team. Prior research has found that the combination of implementation facilitation involving an external facilitator working with an internal facilitator is especially beneficial to sites that are slow to adopt an evidence-based practice [ 41 ]. More broadly, the variability across sites speaks to the importance of using tests of change and ongoing data collection to determine whether implementation facilitation (or other implementation strategies) is having the desired clinical effects, consistent with the principles of a Learning Health System (LHS [ 42 ]). Ensuring the adoption of new clinical practices in healthcare settings is difficult, even with the application of an evidence-based implementation strategy like implementation facilitation—and so ongoing monitoring and adaptation are key.

Limitations

The study findings should be interpreted in light of several limitations. First, the sample size of the study cohort is relatively low, with only eight sites included in this study’s analyses. Of note, site enrollment for the larger study was negatively impacted by the onset and surges of the COVID-19 pandemic. A relative strength of the MMCS approach is that it allows for implementation success analyses both within a site and across multiple sites, but the overall generalizability of the results presented here may be limited due to the small cohort size, which only allowed us to dichotomize the sites into high/medium versus low implementation success categories. Future research should evaluate the use of the MMCS approach on a larger sample of sites with an increased number of implementation success categories to fully understand the impact of factors that can be leveraged to enhance IPV implementation success.

Increased understanding of the influencing factors that impact IPV screening implementation success can inform the tailoring of implementation efforts to allow for successful scale-up of IPV screening implementation in primary care settings. The novel MMCS approach identified key ingredients for the successful implementation of IPV screening programs, including the presence of influencing factors that enable implementation across many domains. This mixed methods in-depth analysis provided nuanced insight into the site-to-site implementation success variability following implementation facilitation efforts as part of a larger clinical trial. IPV screening implementation facilitation efforts that combine resourceful external facilitators with influencing factors that promote understanding of the importance of IPV screening, provide resources attached to the IPV screening tool for screening staff, and involve change makers that drive implementation through consistent engagement with clinic staff members may lead to increased implementation success in primary care settings and beyond.

Availability of data and materials

The datasets generated and/or analyzed during the current study are not publicly available due to privacy or ethical restrictions.

Abbreviations

• Intimate partner violence

Veterans Health Administration

Office of Women’s Health

United States

Matrixed Multiple Case Study

IPV Assistance Program

Implementation facilitation

Information technology

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Acknowledgements

We would like to extend a special thank you to Dr. Bo Kim for sharing her knowledge on the MMCS methodology with us and providing us with expert guidance as we applied the MMCS methodology to this study. We would also like to thank the clinical teams who participated in this initiative.

The views expressed in this article are those of the authors and do not necessarily represent the position or policy of the Department of Veterans Affairs or the United States government.

The research evaluation was funded by the Department of Veterans Affairs, Office of Research and Development, Health Services Research & Development (HSR&D) Services (SDR 18-150: Iverson & Miller). The funder did not have a say in the analyses or content of this manuscript.

Author information

Omonyêlé L. Adjognon and Julianne E. Brady contributed equally to this work and shared the first authorship.

Authors and Affiliations

Center for Healthcare Organization and Implementation Research (CHOIR), VA Boston Healthcare System, Boston, MA, USA

Omonyêlé L. Adjognon, Julianne E. Brady, Kelly Stolzmann & Christopher J. Miller

Women’s Health Sciences Division, National Center for PTSD, VA Boston Healthcare System, Boston, MA, USA

Katherine M. Iverson

Department of Psychiatry, Boston University Chobanian & Avedisian School of Medicine, Boston, MA, USA

Center for Health Equity Research and Promotion (CHERP), Crescenz VA Medical Center, Philadelphia, PA, USA

Melissa E. Dichter

School of Social Work, Temple University, Philadelphia, PA, USA

Massachusetts Veterans Epidemiology Research and Information Center, VA Boston Healthcare System, Boston, MA, USA

Robert A. Lew

Division of General Internal Medicine, Albany Medical College, Albany, NY, USA

Megan R. Gerber

Albany Stratton VA Medical Center, Albany, NY, USA

Pain Research Informatics Multi-morbidity Education (PRIME) Center of Innovation, VA Connecticut Healthcare System, West Haven, CT, USA

Galina A. Portnoy & Sally G. Haskell

Department of Psychiatry, Yale School of Medicine, New Haven, CT, USA

Galina A. Portnoy

VA Palo Alto Healthcare System, Palo Alto, CA, USA

Samina Iqbal

Division of Primary Care and Population Health, Stanford University School of Medicine, Stanford, CA, USA

Office of Women’s Health, Department of Veterans Affairs, Washington, DC, USA

Sally G. Haskell

Department of Internal Medicine, Yale School of Medicine, New Haven, CT, USA

Intimate Partner Violence Assistance Program, Care Management and Social Work Services, Department of Veterans Affairs, Washington, DC, USA

Le Ann E. Bruce

Department of Social Work, Western Kentucky University, Bowling Green, KY, USA

Department of Psychiatry, Harvard Medical School, Boston, USA

Christopher J. Miller

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Contributions

OLA and JEB curated the data, analyzed and interpreted the data, and drafted and revised the manuscript. KMI acquired the funding, conceptualized and designed the study, interpreted the analyses of the data, and substantively revised the manuscript. KS conceptualized the analysis of the data, analyzed the data, interpreted the analyses of the data, and substantively revised the manuscript. RAL conceptualized the analysis of the data and interpreted the analyses of the data. MED conceptualized and designed the study. MRG, GAP, SI, SGH, and LEB conceptualized the study and provided resources for the study’s main activities. CJM acquired the funding, conceptualized and designed the study, interpreted the analyses of the data, and drafted and revised the manuscript. All authors read and approved the final manuscript.

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Correspondence to Omonyêlé L. Adjognon .

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Adjognon, O.L., Brady, J.E., Iverson, K.M. et al. Using the Matrixed Multiple Case Study approach to identify factors affecting the uptake of IPV screening programs following the use of implementation facilitation. Implement Sci Commun 4 , 145 (2023). https://doi.org/10.1186/s43058-023-00528-x

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DOI : https://doi.org/10.1186/s43058-023-00528-x

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The following case study focuses on a 55-year-old male with multiple myeloma and prognosis of undetermined significance. Test your knowledge by reading the background information below and making the proper selection.

A 55-year-old male presents to your office with new symptoms of exertional fatigue. He is otherwise well with no significant past medical history. His hemoglobin is found to be low at 10.6g/dl, with an MCV of 92. He has normal serum ferritin, vitamin B12, and folic acid levels. Absolute neutrophil count is 1.3 x 10 3 /ul and platelets 117 x 10 9 /uL. He has a creatinine of 0.9 mg/dL, calcium of 9.2 mg/dL, and albumin of 3.8 g/dL. A serum protein electrophoresis is performed that demonstrates a monoclonal IgA protein of 1.5 g/dL. A skeletal survey shows occult lytic lesions in the skull and bilateral humeri, and a bone marrow biopsy shows 30 percent involvement by abnormal appearing plasma cells, confirmed by CD138+ immunohistochemical stain.

You make a diagnosis of symptomatic multiple myeloma and review the findings and need for treatment with the patient. The patient wishes to know more about his prognosis and chances of responding to therapy. You explain that you are awaiting a few additional test results that will help you answer his question. Which result would not be considered a poor prognostic feature in this patient with newly diagnosed multiple myeloma?

• (4;14) by FISH
• Beta-2 microglobulin of 7mg/L
• t(11;14) by FISH
• Deletion 13 abnormality by standard karyotype

Explanation

Initial diagnostic work-up for patients with multiple myeloma (MM) should include both conventional karyotyping as well as fluorescence in situ hybridization (FISH) of plasma cells obtained from bone marrow aspiration. FISH should be assessed for poor-risk translocations, including t(4;14) (MMSET translocation; 75 percent of which express FGFR3), t(14;16) (MAF translocation), and deletion 17p (loss of p53). The presence of a chromosome 13 abnormality (monoallelic loss of chromosome 13 or deletion of its long arm 13q) by standard karyotype also confers a poor prognosis. However, when a chromosome 13 abnormality is detected only by FISH the significance is less clear and does not seem to confer the same poor prognosis. It has been suggested that detection of del 13 on karyotype analysis is a surrogate for the proliferative rate of the tumor clone. The t(11;14) translocation, which juxtaposes the cyclin D1 gene with the IgH promoter, is associated with a neutral to favorable prognosis in multiple myeloma. Additionally, a hyperdiploid karyotype also carries a more favorable prognosis in multiple myeloma and is a distinct biologic entity from non-hyperdiploid MM. 1-4

The International Staging System (ISS) for multiple myeloma uses β-2 microglobulin and serum albumin to divide patients into stage I, II, or III disease. A β-2 microglobulin level ≥ 5.5 mg/L would classify a patient as stage III disease, with a median survival of 29 months with conventional chemotherapy (about 80 percent of studied patients) or upfront high-dose chemotherapy and autologous stem cell transplantation (about 20 percent of studied patients). This is compared to a median survival of 62 months for those with stage I disease (β-2 microglobulin < 3.5 mg/L and serum albumin ≥ 3.5 g/dL). 5 Therefore, the ISS remains a powerful and simple non-genetic model to stratify MM patients. However, it should be noted that the ISS prognostic model will need to be validated in the era of novel agents, since drugs such as bortezomib and lenalidomide may overcome the negative predictors in this model.

Currently, there is not a standard recommendation to treat poor-risk patients differently from standard-risk patients, however this is the subject of ongoing clinical studies. It is important to note though that bortezomib appears to ameliorate or eliminate the prognostic significance of these historically poor-risk features (particularly deletion 13, and translocation 4;14) and is generally recommended as part of the induction strategy in such patients. The ability of immunomodulatory drugs such as lenalidomide to overcome poor-risk features has not been as clearly shown to date. 6-8

Note: The true teaching point of this case study is that t(11;14) exists in mantle cell lymphoma and multiple myeloma, and it may identify a possibly good prognostic group.

• Fonseca R, Barlogie B, Bataille R, et al. Genetics and cytogenetics of multiple myeloma: a workshop report . Cancer Res. 2004;64:1546-1558.
• Avet-Loiseau H, Attal M, Moreau P, et al. Genetic abnormalities and survival in multiple myeloma: the experience of the Intergroupe Francophone du Myélome . Blood. 2007;109:3489-3495.
• Chng WJ, Santana-Dávila R, Van Wier SA, et al. Prognostic factors for hyperdiploid-myeloma: effects of chromosome 13 deletions and IgH translocations . Leukemia. 2006;20:807-813.
• Fonseca R, Bergsagel PL, Drach J, et al. International Myeloma Working Group molecular classification of multiple myeloma: spotlight review . Leukemia. 2009;23:2210-2221.
• Greipp PR, San Miguel J, Durie BG, et al. International staging system for multiple myeloma . J Clin Oncol. 2005;23:3412-3420.
• Jagannath S, Richardson PG, Sonneveld P, et al. Bortezomib appears to overcome the poor prognosis conferred by chromosome 13 deletion in phase 2 and 3 trials . Leukemia. 2007;21:151-157.
• San Miguel JF, Schlag R, Khuageva NK, et al. Bortezomib plus melphalan and prednisone for initial treatment of multiple myeloma . N Engl J Med. 2008;359:906-917.
• Kumar SK, Mikhael JR, Buadi FK, et al. Management of newly diagnosed symptomatic multiple myeloma: updated Mayo Stratification of Myeloma and Risk-Adapted Therapy (mSMART) consensus guidelines . Mayo Clin Proc. 2009;84:1095-1110.

Case study submitted by Nicholas Burwick, MD, Hematology/Oncology Fellow, University of Washington, Fred Hutchinson Cancer Research Center.

American Society of Hematology. (1). Case Study: 55-Year-Old Male With Multiple Myeloma and Prognosis of Undetermined Significance. Retrieved from https://www.hematology.org/education/trainees/fellows/case-studies/male-multiple-myeloma .

American Society of Hematology. "Case Study: 55-Year-Old Male With Multiple Myeloma and Prognosis of Undetermined Significance." Hematology.org. https://www.hematology.org/education/trainees/fellows/case-studies/male-multiple-myeloma (label-accessed June 09, 2024).

"American Society of Hematology." Case Study: 55-Year-Old Male With Multiple Myeloma and Prognosis of Undetermined Significance, 09 Jun. 2024 , https://www.hematology.org/education/trainees/fellows/case-studies/male-multiple-myeloma .

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Clinical Presentation: Case History # 1 Ms. C is a 35 year old white female. She came to Neurology Clinic for evaluation of her long-term neurologic complaints. The patient relates that for many years she had noticed some significant changes in neurologic functions, particularly heat intolerance precipitating a stumbling gait and a tendency to fall. Her visual acuity also seemed to change periodically during several years. Two months ago the patient was working very hard and was under a lot of stress. She got sick with a flu and her neurologic condition worsened. At that time, she could not hold objects in her hands, had significant tremors and severe exhaustion. She also had several bad falls. Since that time she had noticed arthralgia on the right and subsequently on the left side of her body. Then, the patient abruptly developed a right hemisensory deficit after several days of work. The MRI scan was performed at that time and revealed a multifocal white matter disease - areas of increased T2 signal in both cerebral hemispheres. Spinal tap was also done which revealed the presence of oligoclonal bands in CSF. Visual evoked response testing was abnormal with slowed conduction in optic nerves.    (Q.1)    (Q. 2)    (Q.3) Today, the patient has multiple problems related to her disease: she remains weak and numb on the right side; she has impaired urinary bladder function which requires multiple voids in the mornings, and nocturia times 3. She became incontinent and now has to wear a pad during the day.   (Q.4)   She also has persistent balance problems with some sensation of spinning, and she is extremely fatigued. REVIEW OF SYSTEMS is also significant for a number of problems related to her suspected MS. The patient has a tendency to aspirate liquids and also solids.    (Q.5)   (Q.6) She complains of tinnitus which is continuous and associated with hearing loss, more prominent on the left. She has decreased finger dexterity and weakness of the hands bilaterally. She also complains of impaired short-term memory and irritability. FAMILY HISTORY is significant for high blood pressure, cancer and heart disease in the immediate family. PERSONAL HISTORY is significant for mumps and chicken pox as a child, and anemia and allergies with hives later in life. She also had a tubal ligation. NEUROLOGIC EXAMINATION: Cranial Nerve II - disks are sharp and of normal color. Funduscopic examination is normal. Cranial Nerves III, IV, VI - no extraocular motor palsy or difficulties with smooth pursuit or saccades are seen. Remainder of the cranial nerve exam is normal except for decreased hearing on the left, and numbness in the right face, which extends down into the entire right side. The Weber test reveals greater conductance to the right. Rinne's test reveals air greater than bone bilaterally.   (Q.7) The palate elevates well. Swallow appears to be intact. Tongue movements are slowed, but tongue power appears to be intact. Motor examination reveals relatively normal strength in the upper extremities throughout. However, rapid alternating movements are decreased in both upper extremities and the patient has dysdiadochokinesia in the left hand.   (Q.8) Mild paraparesis is noted in both legs without severe spasticity. Deep tendon reflexes are +2 and symmetrical in the arms, +3 at the ankles and at the knees. Bilateral extensor toe sign are present. Sensory exam reveals paresthesia on the right to touch and decreased pin sensation on the right diffusely. The patient has mild vibratory sense loss in the distal lower extremities. Romberg's is negative.   (Q.9) Tandem gait is mildly unstable. Ambulation index is 7.0 seconds for 25 feet. (The patient takes 7.0 seconds to walk 25 feet.) Diagnosis: Multiple Sclerosis with laboratory support.   ©   John W.Rose, M.D.,   Maria Houtchens, MSIII,   Sharon G. Lynch, M.D.

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Team effectiveness in academic medical libraries: a multiple case study

Affiliation.

• 1 University of Massachusetts Medical School, 55 Lake Avenue North, Worcester, Massachusetts 01655, USA. [email protected]
• PMID: 16888659
• PMCID: PMC1525325

Objectives: The objective of this study is to apply J. Richard Hackman's framework on team effectiveness to academic medical library settings.

Methods: The study uses a qualitative, multiple case study design, employing interviews and focus groups to examine team effectiveness in three academic medical libraries. Another site was selected as a pilot to validate the research design, field procedures, and methods to be used with the cases. In all, three interviews and twelve focus groups, with approximately seventy-five participants, were conducted at the case study libraries.

Findings: Hackman identified five conditions leading to team effectiveness and three outcomes dimensions that defined effectiveness. The participants in this study identified additional characteristics of effectiveness that focused on enhanced communication, leadership personality and behavior, and relationship building. The study also revealed an additional outcome dimension related to the evolution of teams.

Conclusions: Introducing teams into an organization is not a trivial matter. Hackman's model of effectiveness has implications for designing successful library teams.

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Hackman's framework

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Characteristics of effective teams elucidated…

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Research Article

The use and application of intensive care unit diaries: An instrumental multiple case study

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

Affiliations Intensive Care Department, County Hospital, Region Kalmar County, Kalmar, Sweden, Department of Health and Caring Sciences, Linnaeus University, Kalmar, Sweden

Roles Conceptualization, Formal analysis, Funding acquisition, Investigation, Methodology, Resources, Supervision, Validation, Writing – review & editing

Affiliations Intensive Care Department, County Hospital, Region Kalmar County, Kalmar, Sweden, Department of Health and Caring Sciences, Linnaeus University, Kalmar, Sweden, Research Section, Region Kalmar County, Kalmar, Sweden

Roles Conceptualization, Formal analysis, Methodology, Supervision, Validation, Visualization, Writing – review & editing

Affiliations Department of Health and Caring Sciences, Linnaeus University, Kalmar, Sweden, Swedish Family Care Competence Centre, Kalmar, Sweden

Roles Conceptualization, Formal analysis, Methodology, Supervision, Validation, Writing – review & editing

* E-mail: [email protected]

• Maria Johansson, 
• Ingrid Wåhlin, 
• Lennart Magnusson, 
• Elizabeth Hanson

• Published: February 29, 2024
• https://doi.org/10.1371/journal.pone.0298538
• Reader Comments

Aims and objectives

The study aim was to explore the use of an Intensive Care Unit (ICU) diary within four different ICUs units in Sweden and thereby contribute to practice guidelines regarding the structure, content and use of an ICU diary.

ICU diaries are used to aid psychological recovery among critical care patients, but differences remain in diary writing both within and across countries. Few studies have focused on the combined views and experiences of ICU patients, family members and nursing staff about the use of ICU diaries.

An instrumental multiple case study design was employed.

Three focus groups interviews were carried out with 8 former patients and their family members (n = 5) from the research settings. Individual interviews were carried out with 2 patients, a family member and a nurse respectively. Observations, field notes, documentary analysis and conversations with nursing staff were also conducted. Consolidated criteria for reporting qualitative research (COREQ) was followed.

The qualitative findings firstly consisted of a matrix and descriptive text of the four ICU contexts and current practices. This highlighted that there were similarities regarding the aims and objectives of the diaries. However, differences existed across the case study sites about how the ICU diary was developed and implemented. Namely, the use of photographs and when to commence a diary. Second, a thematic analysis of the qualitative data regarding patients’ and family members’ use of the ICU diary, resulted in four themes: i) the diary was used to take in and fully understand the situation; ii) the diary was an opportunity to assimilate warm, personalised and human care; iii) the diary was used to manage existential issues; and iv) the diary was a tool in daily activities.

Conclusions

Analysis of the instrumental case study data led to the identification of core areas for inclusion in ICU diary practice guidelines.

Citation: Johansson M, Wåhlin I, Magnusson L, Hanson E (2024) The use and application of intensive care unit diaries: An instrumental multiple case study. PLoS ONE 19(2): e0298538. https://doi.org/10.1371/journal.pone.0298538

Editor: Sascha Köpke, University Hospital Cologne: Uniklinik Koln, GERMANY

Received: April 20, 2023; Accepted: January 25, 2024; Published: February 29, 2024

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

Data Availability: Data cannot be shared publicly because of the risk of identifying individual study participants. Data requests may be sent to the head of research at Region Kalmar County, Professor Cecilia Fagerström: [email protected] .

Funding: The first author was partly financed for her PhD education of which this study forms a part by the Health Research Council of South East Sweden and Kalmar County Council. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript".

Competing interests: The authors have declared that no competing interests exist.

Introduction

Intensive Care Unit (ICU) diaries are increasingly being used in intensive and critical care nursing practice as an intervention that may facilitate the patient’s psychological recovery. Namely, by describing what happened to them in the ICU and about their health condition [ 1 – 3 ] with the aim of helping to put possible unpleasant memories into context [ 4 ]. The authoring of diaries may help both patients and family members process the critical illness experience during and after the time in the ICU [ 1 , 5 – 7 ] as well as support family members’ bereavement process when the patient does not survive [ 6 – 8 ]. Due to the COVID 19 pandemic, some ICUs opened up fewer diaries because of worries about infection control and due to excessive workload. In addition, visiting restrictions limited the use of diaries written by family members [ 9 ]. However, web-based solutions are under development and may in the future become a replacement for or supplement to hard copy diaries [ 10 ]. A hard copy diary is described as a handwritten booklet written in collaboration with nurses/staff members and family to the unconscious patient based on a chronology of clinical and social events that occurred during the ICU stay [ 6 ]).

Qualitative studies have highlighted how the written content in the diaries helped patients to realize what they had undergone, provided a sense of coherence and understanding after a critical illness [ 11 – 13 ] and acted as a support in the process of setting up realistic goals for recovery [ 7 , 11 , 14 ]. According to Backman & Walther [ 6 ] if photos are added, they might augment the information and help to make sense of the information provided, akin to a reality check. The diaries could also convey a feeling of humanised care despite being immersed in a technological environment [ 15 ].

Earlier quantitative studies have revealed how the diaries reduced the occurrence of patient post-traumatic stress, depression, and anxiety related to critical illness and critical care [ 16 , 17 ] as well as aid the wellbeing of their relatives [ 18 , 19 ] and improved patient health-related quality of life after a critical illness [ 20 ].Overall, the quantitative studies have been criticised due to small sample sizes and differences in the diary’s application [ 21 , 22 ]. Thus, it remains unclear as to the efficacy of ICU diaries in reducing PTSD, anxiety and depresession in patients and family members [ 9 ]. The majority of previous studies consist of mainly qualitative studies, which highlight that the diary helped to fill in the memory gap of the patients [ 6 , 7 , 23 ]. Also, the diaries assisted the patient when working through their ICU experiences [ 14 , 24 ] and at the same time provided individual or improved quality care [ 14 , 24 ]. Taken together, considerable diversities remain in diary writing both within and between countries which make it difficult to draw comparisons among the existing empirical studies. A major identified barrier regarding the use of ICU diaries for health care professionals is lack of dedicated time, which was further exacerbated during the COVID-19 pandemic [ 9 ]. A consensus exists on the need for guidelines to support diary writing for use in current critical care nursing practice [ 25 – 27 ]. In this regard, the international ICU diary network (established in 2012 by a group of ICU nurses as a non-profit organisation) acts as a vehicle to help with the implementation of ICU diaries via coordinating information, projects and new empirical studies for interested health care staff [ 28 ].

The overall aim is to explore the use of the ICU diary within four different ICU units in Sweden, and thereby contribute to practice guidelines regarding its structure, content and use.

The research questions were as follows:

• What are the current practices surrounding the use of a diary in the chosen ICU settings? What local guidelines exist (if any) and in what ways (if any) are they implemented in clinical practice?
• What strengths or positive aspects are identified by nursing staff, former patients, and family members of ICU patients with regards to current practices in the chosen research settings concerning the use of an ICU diary?
• Further, what are the challenges or drawbacks that are identified by nursing staff, former patients and family members of ICU patients with regards to current practices in the chosen research settings concerning the use of an ICU diary?
• What are the core areas for consideration arising from this multiple case study that can be taken up in future development work concerning national clinical practice guidelines on the use of the ICU diary?

An instrumental multiple case study design was adopted for the study. An instrumental case study enables a contemporary phenomenon to be investigated in-depth and in its real-world context–in this instance, the use of ICU diaries. Further, a multiple case study approach was chosen as it enabled the researchers to explore similarities and differences in the individuals’ experiences within cases and compare and contrast the experiences across cases [ 29 ]. A case study design provides opportunities to use multiple data sources to answer ‘how’ and ‘why’ questions- in this case, about ICU diary use, implementation and how to further develop the diary. The use of several data sources may also enhance data credibility to reach a holistic understanding of the phenomenon [ 29 , 30 ]. The use of evidence from multiple sources also means a kind of triangulation, where the findings have been supported by more than a single source of evidence [ 28 ]. This study followed Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups [ 31 ] (See S1 Table ).

Study setting

There are approximately 41070 admissions to 82 adult intensive care units in Sweden annually [ 32 ]. ICUs are situated in university, county and district hospitals. County and district hospitals have general ICUs. The ICUs in university hospitals may have a range of specialized ICUs, for example; burns ICU, neurosurgical ICU, medical ICU, surgery ICU and thoracic surgical ICU. The specialized ICUs serve other hospitals in their region when specialized care is required. The study took place in 2018 between March and October at four different Swedish ICUs (units A, B, C and D) in four ICU settings, equal to four cases where the ICU diary has been developed and implemented. Henceforth, ICU settings are called cases, which means everything associated with the settings, such as documents, observations, notes, routines, interviews, and study participants. One ICU setting was a university hospital, and three were county hospitals (See Table 1 ).

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https://doi.org/10.1371/journal.pone.0298538.t001

The included cases were purposively selected as they were representative of a diversity of care provision, context, and implementation practices of the ICU diary [ 29 ]. All participant hospitals were in the same geographical region of Sweden which covers an approximate radius of 170 km by 46954 km 2 , and they all formed part of a regional research collaborative initiative financed by the involved health care regions to stimulate closer cooperation concerning health care research. Nursing staff consisted of specialist critical care nurses with a bachelor level degree and assistant nurses. The staff-patient ratio at the time of the study was between 1.0 and 1.2. Medical staff consisted of 1–2 chief intensive care specialists and several junior physicians and at the units there were 1–2 head nurses of the department. There were no psychologists attached to the ICU units. Nurses, physicians, and nurse assistants provided psychological support to patients and their family members. ICU diaries have been used at the participating units for between 25–30 years.

Data collection

A protocol for conducting a multiple case study was established (See S2 Table ) in accordance with Yin’s (29). The protocol was a procedural guide for collecting data for a case, including a set of field questions to be addressed by the researcher (first author, MJ). The role of the protocol was also a standardised agenda for the researcher’s line of inquiry to increase reliability [ 29 ]. The data sources included observations, field notes, documents, informal, unstructured field interviews, a telephone interview, individual interviews, focus group interviews and a text message. A trusted external secretary with prior experience as a medical secretary transcribed the recordings.

Observations.

The first author (MJ) engaged in direct observations of selected cases for approximately 24–32 hours respectively in the ICUs (B, C, D). MJ was working clinically at that time in ICU A, and was subsequently familiar with the unit thus needed no assistance in collecting documents.

Field notes.

Field notes consisted of a continuous record of responses reported from the different units according to the study protocol. They were written by MJ both in the ICU but also directly after each observation period.

Collected documents included pamphlets related to the patient in critical care, documents about patient diary guidelines and informed consent forms about diary writing/photography.

Informal field interviews.

The field interviews took the form of spontaneous, informal conversations which described the nursing staff’s views and experiences of how the ICU diary was used within everyday nursing practice on their respective unit. These informal interviews were not audio-taped but rather written up in the form of field notes by the first author (MJ).

Individual interviews.

An interview was conducted with one patient and their family members recruited via telephone from the ICU follow-up programme at one of the ICU settings. The interview was based on the questions from the study protocol but reformulated into open-ended questions ( S2 Table ). Moreover, an interview with a nurse with experience of developing diary implementation in Sweden was carried out in the same way. The interviews were audio-taped with the participants’ permission, ranging from 60–70 minutes in duration and subsequently transcribed verbatim. These participants were originally invited to the focus group interviews but were prevented from attending for various reasons. Nevertheless, they wished to communicate their experience of using the diary. A telephone interview/conversation and a textual message from a former patient were also included in the data. Field notes were duly written directly after the telephone conversation had taken place.

Focus group interviews.

Three focus group interviews with a total of 8 patients and 5 family members at the participant hospitals were conducted to explore the use of an ICU diary in the respective care settings. The design of the ICU diary was developed in keeping with each unit’s guidelines and practices. The participants were only aware of the version they had received from their respective ICU. Nurses from the individual ICU follow-up programme recruited the participants via telephone. Information about the focus group sample is provided in S3 Table .

Patients had been discharged for a period of seven to fourteen months depending on their recovery process and were invited to bring family members with them to the focus group session. The inclusion criteria for patients and family members included; being a former ICU patient, having an ICU diary, being willing to share their experiences and aged 18 years or older.

In accordance with Krueger & Casey [ 33 ], open-ended questions were posed in order to allow for explanations, descriptions and illustrations relating to ICU diary writing and practices. Key questions included the following; how was it to read/write in the diary? When was the diary handed over? When were the photos handed over? What are your thoughts about the content in the diary? MJ moderated all focus group interviews. The second author (IW), a critical care nurse specialist with a PhD in caring sciences, operated as an assistant moderator and concluded the interview by briefly summarizing the main points and asked if the summary reflected what participants had experienced in the group [ 33 ]. Interviews were audiotaped, and were of 86–127 minutes in duration. The data were transcribed verbatim. Unstructured field notes were written by MJ directly after the interviews to document key remarks and reflections, such as something surprising and/or unexpected to MJ or how a group’s responses were similar to or different from earlier focus group sessions [ 33 ].

Data analysis

In keeping with Yin’s [ 29 ] recommendation with case study analysis work, the authors duly returned to the core research questions. To help answer the first research question, the extensive amount of practical information from the observational studies, documents and informal, and unstructured field interviews were systematised with the help of a matrix (see Table 2 below) developed from the study protocol (See S2 Table ) [ 29 ].

https://doi.org/10.1371/journal.pone.0298538.t002

Second, to answer the remaining research questions (2, 3 and 4), a thematic analysis, inspired by Braun and Clark [ 34 ], was conducted of the qualitative data from the focus group interviews, individual interviews, telephone interview, and textual message [ 34 ]. This approach was deemed suitable for identifying, reporting and interpreting patterns of meaning within the transcribed data. It was also chosen for being a flexible yet rigorous method for examining the perspectives of different research participants, highlighting similarities and differences and also generating unanticipated insights [ 34 ].

The analysis commenced with repeated reading to be familiar with all aspects of the data. Thereafter, a coding process began using highlighters and writing notes on the texts. Extracts that demonstrated each code were brought together in Word documents, one for each pattern. An extract might be uncoded, coded once, or coded many times as it was relevant. This way, all the codes with their extracts were organised in their document. The documents were organised into theme piles. In the final step of the analysis, core themes were developed that reflected patients’ and family members’ use of the ICU diary (See Fig 1 below).

https://doi.org/10.1371/journal.pone.0298538.g001

Ethical considerations

The study was conducted in compliance with the established ethical guidelines of the Declaration of Helsinki [ 35 ] and the Regional Ethical Committee in Linköping, Sweden provided formal ethical approval for the study (Dnr 2017/550-31). The clinical medical director at each of the respective units was informed about the study and approved the study. Potential focus group participants were recruited through the respective ICU’s follow-up services, and the hospital with the strategically optimal location was chosen for the interviews. Participants were financially compensated for their travel costs.

Information about the study was repeated verbally to participants prior to the start of the observation study and the interviews. Voluntary participation, informed consent, confidential treatment of all data and the right of participants to withdraw at any time without repercussion to their care or working situation (as appropriate) and appropriate storage of the qualitative data were all emphasised. Informed oral consent by the observation participants was witnessed by the first author and consisted of the participants verbally stating that they agreed to the first author observing activities and interactions involving the participant as part of her research study. Informed oral consent by the interview participants was also witnessed by the first author and consisted of the interview participants verbally agreeing to take part in an interview as part of the first author’s research study. Verbal consents were documented by the first author as part of her methodological field notes and as approved by the regional ethics committee. As the first author was working as a critical care nurse in ICU A, qualitative interviews were not carried out in this particular setting [ 36 ].

Table 2 below provides an overview matrix of the main similarities and differences regarding the structure, content, and ICU diary use within participating ICU settings arising from the systematized observational study, documentary and informal field interview data as outlined above. The matrix is supplemented with a written summary description of the four ICU contexts and main current practices drawn from the qualitative data. Core findings from a thematic analysis of the qualitative data are subsequently presented.

ICU contexts

It was self-evident by nursing staff in all ICUs to involve family members in writing/reading the diary without them feeling obliged to write. Family members might come and go around the clock at some units, but in others, staff agreed on their visiting times. ICU nursing staff had diverse experiences of diary writing from 10 to 23 years. Prepared diaries were readily available on the ICUs in a room intended for follow-up services.

Staff from the general ICUs’ follow-up services had regularly networked since 2012, together with another two ICUs in the region in order to exchange experiences about diary implementation and follow-up services. At that time, all participant ICUs had the same members in the follow-up services and in the diary group who prepared and were responsible for diary implementation.

Nursing staff at each ICU had established their own purpose statement in the absence of national guidelines on patient diaries. Diary writing had been realised as a spontaneous initiative by nursing staff, and the activity had advanced often through “learning by doing”. The assumption being that the patient needed to know and come to terms with what happened while they were in ICU. Also, family members needed help to also understand and process the patient’s stay.

Written guidelines were found in one ICU and comprised inclusion, exclusion criteria, writing style, informed consent, the process around photographing, and handover of the diaries. Another unit was developing written guidelines, but the other units had only oral guidelines about the structure, content, and use of the diary. Experienced nursing staff communicated informal guidelines to new employees and less experienced ICU nursing staff. The absence of clear, written guidelines meant that many patients did not receive a diary because of the hesitation in deciding which patients should get a diary and how to prioritise the process. The uncertainty was expressed as a cause of frustration among the participant nursing staff.

Diary structure.

The diary was an A5 notebook, lined, laminated cardboard folder, and spiral bounded or bound. Some ICU nursing staff had chosen a picture of a well-known sight from the town to act as the cover. Others had chosen colourful images such as flowers or trees. The ICU diaries’ format was open, only blank pages, handwritten, and without any editing (that is, there was no one responsible who approved the diary before it was handed over to the patient). On the initial page/s, the diary included dedication to the patient and the patient’s name.

Next, diaries usually contained a summary and a reason for the patient’s admission to ICU and daily entries. The summary and closing framed the diary’s story about the time in ICU. There was also a glossary, and in three of four ICUs, generic photos on the most common ICU medical equipment.

Diary content

All diaries contained a page called ‘Who am I? What is important for me?’ The page asked for the patient’s sleep habits, favourite sports, favourite food and drinks, music, and philosophy on life which family members were requested to complete. The diaries in some ICUs comprised the 24-hour circle in the ICU with an associated timeframe. This information highlighted that the activities varied around the clock and explained that somebody was always nearby the patient. Next followed the narratives of daily activities with and around the critically ill patient, which formed a central part of the diary. The narratives contained information about medical treatment, daily care of the patient, information about progression, such as mobilising out of bed, conversely how the patient’s condition had deteriorated, and that the patient was approaching death.

Other entries were psychosocial, which expressed and confirmed the presence of the family members at the bedside. Family members added information about what happened at home or greetings from other family members. Entries included reflections around the weather, seasons, Swedish festivals, sports events together with entries of a more personal nature. Staff members who wrote the entry included the whole team, dated, and signed the note. A unique contribution from staff at one ICU was a poem at the end of the diary intended to provide a respectful content if diary entries were sparse.

Use of the diary

All units kept the diaries at the patient’s bedside so that nursing staff and family members could write whenever they wanted to. The diary was seen as the patient’s property and was not deemed to be part of the patient’s medical records. Written or oral guidelines stated that a diary should be offered to the patient when they were expected to be cared for >72 hours or for those who ‘needed’ it. In one ICU, there was an ambition to open up a diary to all patients admitted to the ICU, especially patients cared for following a cardiac arrest, which meant a shorter care stay than three days. Care of patients with cardiac arrests denoted heavy sedation, ventilation and connection to a range of cannulas and tubes for about 24–36 hours and then awoken. In general, this group were deemed to benefit from a diary.

The nursing staff did not open an ICU diary for patients with severe brain injuries, and dementia, poor prognosis nor for patients who did not speak/understand Swedish. Often, some nursing staff commenced a diary immediately when a patient was admitted to ICU, but sometimes days elapsed, and the diary writing did not take place. Reason/s for the delay included lack of time or difficulties to start the diary as the summary was deemed to be the most essential and extensive aspect. In some units, assistant nurses frequently opened a diary as they had more time to initiate the process than the registered nurses.

Two ICUs kept registers about patients who had a diary. This included a checklist with the patient’s name and informed consent given by family members to photograph the patient. The checklist was stored in a diary file in the staff room and accessed by nursing staff who belonged to the diary group. Other ICUs asked family members for informed consent orally in conjunction with information about the diary without any written documentation. The consensus was that the photographing was a part of the treatment and therefore did not necessitate permission from the patient or family members, thereby facilitating the prompt starting of a diary and with photographs. However, the patient was required to give retrospective consent once they were sufficiently well enough to receive the diary.

Use of photographs

All staff from the four ICUs used photos to augment the written content. Nursing staff were aware of the photos’ supportive role and that the patients frequently requested more pictures. Usually, the first photographs were taken when the patient was fully sedated, ventilated, and linked to a range of tubes and cannulas. Subsequent photos were taken on significant events and progressions during the ICU stay. Typically, the patient was photographed at a distance so as not to expose the patient at an inappropriate angle.

However, nursing staff at one ICU meant that the pictures should be realistic, and nursing staff should go close to the patient when photographing because the patient needed to recognise themself. The photos should assist as a ‘reality check’ when setting goals for recovery.

Three ICUs used a digital camera with memory, where nursing staff might print out the photograph straightaway and then delete the original from the memory card. The fourth ICU moved and stored the pictures in image management on a desktop. At one ICU, the pictures were pasted in the diary immediately, close to the written text, so that text and image interacted. Sometimes, photos were collected in an envelope at the back of the diary.

Another ICU used mounted the photos directly in their context in the diary but left the other side empty. This made it possible for the patient to remove the picture but without destroying any existing text.

In contrast, two ICUs did not include the photos directly in the diaries. The rationale being that the patient needed a ‘face to face’ meeting with ICU nursing staff and an explanation of the photos to fully understand the pictures and be allowed to ask any questions. Otherwise, they feared that the patient might be frightened or re-traumatised if no explanations was given. Another reason for the personal meeting was to obtain retrospective consent for the pictures. If the patient refused the photos, they were destroyed.

Handover process

The diary followed the patient by the nursing staff placing the diary at the end of the bed or giving it to family members on transfer to the general ward. Diary group/follow-up service staff visited the patient at the ward when they were transferred from the ICU. During this visit, the photographs were discussed in those cases they were not included in the diary. If the patient wanted the photos at that time, they were handed over. Otherwise, the pictures were discussed at the follow-up service later and then handed over. Patients who did not attend any follow up service received their photos by mail at home, if they requested them.Family members where the patient did not survive, received a telephone call about 6–8 weeks after the patient’ death and were invited to the follow-up service to discuss the patient’s stay at the ICU.

Thematic analysis findings

Four interconnected themes were derived from the qualitative thematic analysis that reflected patients’ and family members’ use of the ICU diary. The themes are represented in the following sections and highlighted in Fig 1 below. Participants are cited using the number of the focus group and P (atient), S (pouse) number identifier.

The diary was used to take in and fully understand the situation.

The chronology in the diary and the everyday language were understood as valuable characteristics because family members expressed feeling unsure about specific dates and events during their family member’s ICU stay. Even in the diary, patients and family members sometimes had difficulty distinguishing specific dates. A positive aspect of the ICU diary highlighted by participants was the ability to read and re-read the information to assimilate it.

That you can go and look in the diary a little now and then and think about it ( I/S1)

Patients and family members requested ongoing summaries in the diary, as the information was sometimes perceived to be insufficient or irregularly provided, especially during more extended periods of stay. The diary was mainly experienced as a tool to help process the time in ICU by describing what daily life looked like, which was not included in the medical records.

Patients described dreams, nightmares and unreal experiences that caused long-standing problems post-ICU discharge despite a few other more positive memories. This meant that patients and family members had different experiences of events which sometimes led to misunderstandings among families. However, patients expressed missing entries with sufficient personal details which described confused thoughts they had expressed and what the thoughts were about, so they might orientate themselves. Patients expressed a need to know that these confused thoughts were often of a frequent occurrence.

The nursing staff explained to my wife and children–but nothing in the diary , about the confused thoughts . They said it calmly , and that he’s going to recognize you . ( II/P3) .

All patients and family members emphasized how important the summary was, which told the story prior to admission and why the ICU admission had taken place. Despite this, patients and their family members experienced that critical and life-threatening episodes were sometimes neglected, and they wanted more realistic adjectives to be used such as “deadly condition” and “hovering between life and death” to understand how critically ill the patient was. Another drawback relating to missing information was the setting’s description with its sounds, sights, machines and bed. In other words, former patients wanted to have events clarified, which were known to be the source of nightmares, distorted thoughts and hallucinations; as an example, the ICU bed was often experienced as a container, boat or a railway truck. The ICU diary became a way of summarizing and making countless details visible in a single concrete picture. It was considered as a reliable document against which to check their personal memories of their ICU stay.

The diary is fantastic; without it , I would not have known anything . The wife has a hard time reading the diary . The children also find it difficult . ( I/P2) .

The photographs were expressed by patients as being personal and they offered them a window into the past to fully understand their history. All insisted that it was important that photos were tightly connected to the written texts. Some had received photos in bulk (in envelopes) connected with the follow-up services, to insert them independently in the diary.

I had a need to get a whole of the ICU stay . I got loose pictures that I could not put into context ( telephone interview)

In general, patients wanted realistic photographs without embellishments because they wanted to know their actual health status in order to set realistic rehabilitation goals. They also requested close-ups to be able to recognize themselves, otherwise, they argued that it could be anyone in the photo. Patients with realistic pictures of themselves with all the equipment, expressed that it did not feel so scary anymore.

Yes , but then that’s not pleasant , you see it’s me , but further away , then it wouldn’t have been easy to see that it was me ( II/P2) .

The diary was an opportunity to assimilate the warmth, personalized and human care.

Patients and family members found the diary to be personally touching. They explained that naming the patient in each entry gave identity and a personal touch to the diary. They found an unexpected kindness in the staff’s diary writing which deeply moved them. The diary was thoughtful and often well-written with warmth that patients expressed that they would carry with them for life.

They (nursing staff) really showed from the bottom of their hearts that they liked me , that I’m important ( II/P2) .

The diary bore traces of the authors’ varying ability to express themselves, which participant patients and family members appreciated. Family members felt that the nursing staff was there for them all the time, even if it was for the patient’s sake. The diary testified that the patient was cared for with respect and not as an object. For family members, the diary was considered a symbol of human care and hope in situations where the outcome for the patient was uncertain during the ICU stay.

We can read in the diary . ‘We see a slight improvement’ . This little word is so important ( I/S2)

Some former patients proudly read descriptions such as “the ward’s trump card”, “the ward’s sunshine story”, which further underscored the personalized caring and how sick the patient had been and survived the trauma. Others read about how the nursing staff washed the hair of unconscious patients, helped with the first shower, provided offers of TV watching and fulfilled requests for particular drinks.

I think it’s nice to read . Today you have sat on the edge of the bed , and it’s the nursing staff who wrote . They (nursing staff) care so much about one . It warms . ( I P3) .

Patients emphasized the importance of the family members’ notes. However, many family members admitted that they could not write in the diary themselves. They expressed that they were too sad and worried, but they experienced it was good to read the nursing staff’s entries as they found it to be sign of hope among all the sorrow. Patients felt that family members had been well-taken care of. Patients and family members expressed that the support and empathy that they could make out in the diary acted as a long-standing form of support for them.

The diary was used to manage existential issues.

Spiritual issues about the meaning of life emerged for patients experiencing a life-threatening illness, especially when the trauma was lived through and vividly described in the diary. Some of the patients had experienced a near death experience on a visit related to cardiac arrest and asked themselves. Why did I get sick? Why did I survive? The patients might go back and look in the diary from time to time and tend to ask themselves the same existential questions. Patients admitted that it was often serious issues that were raised and that were difficult to share with “outsiders”. Patients and family members believed that one must have been there to fully understand. The pictures in the diary might explain the problematic situation and the ever-present threat of death. They explained that the diary “sharpened the mind”, to pay intention to how fragile life could be and how quickly it could change.

However , it was strange to me; why did I get this disease . I kept thinking , why did I have a cardiac arrest ? For twenty years I haven´t had anything , but suddenly I got it ( II/P2) .

Family members admitted that it caused a disturbance in the family, when a member was ill with a life-threatening illness but that the family was brought together by reading the diary. Some patients noted that they found it challenging yet exciting to read what they had been through in the diary and experienced gratitude that they were alive and believed that life was now too short to dwell on existential issues. Patients admitted that they were not afraid of the future and felt that the experience had empowered them in a way as they now appreciated life even more. Even for family members, existential issues could deeply touch them as illustrated by the following quotation.

Mum , where do you have the diary ? So , she can crawl away for herself , so she is 27 , and then I see how a few tears fall , and then she comes and holds me , says mother , I’m so glad you’re here ( II/P3) .

The diary was used as a tool in daily activities.

Family members described the diary as a tool for information and communication during the ICU stay. To access, understand and take in the medical information. They found the diary glossary to be useful as it often explained the use of medical terms, which eased conversations with nursing staff and provided a feeling of being included. Family members described the diary as available when needed and that they used the diary as a company, comfort and sometimes as an intimate friend at the ICU.

It was great to not just sit alone in the waiting room spending time and biting nails . It was great to have a diary available ( III/S1) .

The diary was viewed by family members as something to keep them occupied with instead of constantly following the monitoring screens and as a way of overcoming uncertainty. Family admitted that they wrote not only for the sick person but also for their own sake to unload their personal worries and thoughts.

On discharge home from hospital, participants explained that the diary became a basis for the patient to ask questions. The diary also became a support for family members to be able to account for the course of events. The diary was perceived as a reliable document about the time spent in ICU. Pictures of family members showed that they had been there during the critical illness.

All visits are listed here . I think it is vital that all the sons are in the pictures and not just S and I ( I/S2) .

However, the diary could evoke unpleasant memories for some patients, which caused the diary to be set aside.

I experienced the diary as a bit silly initially , but now I can get hurt by it when I look at it; the trauma remains and gets even more challenging . (I/P2)

A participant former patient had used the pictures in the diary as proof of how ill they had been in their effort to convince persons in authority. Others had let family members and friends read the diary themselves, as they had not been able to tell and re-tell their story.

The aim of the study was to explore the use of the ICU diary within four different ICUs units in Sweden and thereby contribute to practice guidelines regarding its structure, content and use. The practice recommendations arising from the study findings can be summarised into three areas, namely the need for guidelines, guidelines outlining the content of the diary, and how the diary making process reflected the approach of person-centred care. Each will be considered in turn. This is followed by a discussion of the study’s main methodological considerations.

The need for practical guidelines

The study revealed that it was left to the discretion of the nurse caring for the patient if a diary was opened up. The rather haphazard selection of patients for keeping a diary has previously been demonstrated in Scandinavian surveys 1 [ 4 , 24 ]. National guidelines could act as a support for priority setting and to provide guidance as to whom and when to open up an ICU diary. A common standard would make it possible to follow up on the diary as a caring intervention continuously and systematically. National guidelines may pay attention to the diary making process and so increase awareness among nursing staff that the diary can act as a useful debriefing tool and as a help in reorientation for the patients [ 1 , 2 , 17 , 23 ].

Based on the study findings, family members’ roles in the ICU setting are important to highlight in the national guidelines, including the promotion and support of their active participation in the writing, including the start-up of the diary. It is known from previous studies that family members benefit from the diary [ 5 – 7 ] and that the diary may also act as bereavement support if the patient should die [ 8 ].

In the current study, patients with severe critical illness, (little chance of survival), dementia, and people with learning disabilities were automatically excluded. Therefore, guidelines need to outline that the diary may also benefit family members and then the green light is given to commence a diary to broader groups of patients. Further, it is regarded as a human right to be provided equal treatment as a Swedish inhabitant, and this approach would also include patients with other languages [ 37 ]. The diary may be translated in some way and/or compiled with more pictures and generic pictures.

The study highlighted that guidelines need to be flexible, only stating certain main principles and being considered for every person admitted to the ICU. Further, it showed how practical knowledge sustained the writing as patient categories cared for shorter periods than three days were included. An example is patients having had a cardiac arrest, who were deemed to need follow-up support as this group of patients tended to develop both cognitive and emotional difficulties [ 38 ].

Guidelines outlining the content of the diary

The study revealed that patients and their families requested relevant, realistic content with close-up pictures in the diary because they wanted to know the main details to gain a better understanding of the patient’s illness and what happened to them during their ICU stay. The findings were contradictory to those of the nursing staff, in keeping with a previous study that demonstrated how the staff weighed every word to avoid harming the patient in any way [ 39 ]. It has been previously noted how nursing staff experienced diary writing as “complicated in its simplicity”, due to difficulties finding “the right words” in writing and at the same time providing comfort and confidence that patients and family members needed both during and post the ICU stay [ 40 ]. Further, it was noted that pictures were often taken at a distance and avoided being taken of a patient with a swollen face, which is in line with previous findings [ 14 , 26 ]. This poses the question of whether nursing staff are overly cautious with regards to diary content to avoid causing the patient any potential offence or distress. In addition to the diary content.

The process of photographing highlighted the main differences between the four cases and were related to local legal and ethical considerations. Previous research highlighted that photography caused additional considerations and units removed pictures in the diaries that were taken without prior consent and that could potentially impinge on patient privacy [ 12 , 14 ]. On the other hand, patients who were provided photographs appreciated them and wanted additional ones [ 41 ]. Previous studies have demonstrated the value of pictures noting that photographs added realism and neutralized frightening fantasies [ 42 ] and that the diary with its text and photos can be seen to be important to induce post-experience reflections [ 43 ].

How the diary making process reflected the approach of person-centred care

The participant family members in the study underscored how they were naturally offered to participate actively in care by reading/writing in the diary. In this way, they experienced themselves as valuable and equal partners. Writing the diaries was about doing an intervention with patients and their families in partnership rather than ‘to’ them. Family members highlighted the personality of the critically ill patient, and together with the nursing staff, they helped to meet the patient’s needs for information and support. Writing a personal diary is in line with current healthcare policy concerning people-centred care, which means putting patients and their families at the centre of decisions and working alongside them to secure the best possible outcome [ 44 ]. Establishing a partnership in the ICU was challenging because the patient was not awake and unable to tell their life story. However, the diary and the page with questions ‘Who am I? What is important to me? were created to understand the patient as a human being with physical, psychological, and existential needs. This information was often captured from family members and used to tailor conversation topics, activities and motivational features in daily nursing care. Likewise, the diary itself may be regarded as documentation of a piece of life that would otherwise have been unknown to the patient.

Patients expressed feeling cared for when they read a diary written primarily for them as the diaries were dedicated to the patient. The writing helped to maintain a feeling of togetherness among the family members and nursing staff, which has been expressed in previous studies [ 8 , 45 , 46 ]. The belief that diaries are a caring activity was coined by Roulin, Hurst & Spirig [ 47 ] as writers from different backgrounds took part in an everyday activity for the patient’s benefit. A personal diary with personal photographs also demonstrates that the nursing staff has been willing to go that extra mile for the patient [ 12 ]. Diary writing also created a caring relationship between the patient, family members and nursing staff [ 48 ].

Methodological considerations

The study was carried out by using a qualitative multiple case study methodology, where the study protocol acted as a guide helping the first author in carrying out the data collection from a single case as one of several in the multiple-case study approach [ 29 ]. It can also be seen as a way of increasing the reliability of the case study research and a possibility to replicate findings across cases. Multiple methods of data collection, for example, the combination of focus groups, the individual interviews and notes from observational studies were used to identify similar, different, or complementary views and experiences across the different participant groups. The approach allowed for methodological triangulation and increased the likelihood that findings were credible [ 29 , 30 ].

Limitations.

A study limitation is that the four ICU settings were located in one geographical area of Sweden so that the degree of transferability of the findings to other settings can be questioned. Nevertheless, the study likely reflects the different use of ICU diaries in Sweden. Further, it can be argued that the findings are applicable to other ICU settings with similar publicly financed health care systems. The study can be considered as a first step in a longer-term research and practice development work towards national guidelines regarding the use of the ICU diary.

The first author (MJ) was an ICU nurse at one of the participating clinics, so it could be argued that she potentially lacked objectivity and was biased in her data collection and analysis activities due to her insider knowledge of ICU A [ 36 ]. Nevertheless, MJ kept detailed field notes which included reflections of her researcher role in the settings which were duly discussed on a regular basis with her doctoral supervisors. Also, it can be argued that MJ’s insider knowledge was an advantage as she was familiar with the ICU context so that she was also able to ask relevant questions and was able to have an in-depth understanding of participants’ responses and actions.

Based on the findings it can be argued that clinical practice guidelines concerning ICU diaries would help to ensure their wider and more consistent use for all ICU patients, as opposed to a more selective groups of ICU patients. Second, they would help to make the role of family members more transparent and valued. Finally, they would provide concrete guidance to nursing staff about how best to write in the ICU diary to ensure relevant, real-world content bringing the human element and aiding with genuine person-centered care.

Relevance to clinical practice

The findings are intended to act as an impetus for future practice development work concerning ICU diary guidelines in ICU settings in other health care regions in Sweden and internationally in countries with a similar health care system to that of Sweden. The aim being to help facilitate and promote nursing staff’s everyday clinical practices concerning ICU diary writing in keeping with a person-centred approach.

Supporting information

S1 table. consolidated criteria for reporting qualitative research (coreq): a 32-item checklist for interviews and focus groups..

https://doi.org/10.1371/journal.pone.0298538.s001

S2 Table. Study protocol for instrumental multiple case study.

https://doi.org/10.1371/journal.pone.0298538.s002

S3 Table. Description of focus group interview participants.

https://doi.org/10.1371/journal.pone.0298538.s003

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Generation of medical case-based multiple-choice questions.

1. Introduction

1.1. historical background, 1.2. structure of a case-based mcq.

• The stem (sometimes the portion referred to as the “question”). This might consist of a simple question, but might also be more complex, and include a scenario and media. The key element in creating a robust multiple-choice question is to ensure that the stem is well-defined and focused. The stem of the query must contain the primary concept.
• Alternatives (sometime referred to as “options”). These include all the items, from which the user must select one.
• Answer (sometimes referred to as the “correct answer” or the “key”). This is one of the alternatives, and is the actual required answer to the question. The crucial characteristic is that the selected option deemed as accurate must be absolutely indisputable, without any doubt or debate. It is preferable to have a manuscript citation or reference on hand for verification purposes. When providing a correct answer, beware of using ambiguous phrases like “frequently”, “often”, “rarely”, or “sometimes”. These hints indicate that an answer is correct and demonstrate test-taking intelligence rather than subject content knowledge.
• Distractors. There are all the alternatives that are not the answer. From a cognitive perspective, it is acceptable to have two distractors. However, in health sciences testing, it is more common to have three or four distractors. Writing plausible distractors can be the most difficult aspect of developing a well-formulated examination.

1.3. Transition to Clinical Significance

1.4. emergence of case-based mcqs, 1.5. integration of technology, 2. the significance of case-based mcqs, 2.1. the integration of theory and practice, 2.2. evaluating higher-order cognitive abilities, 2.3. improving clinical readiness, 2.4. embracing contemporary pedagogical approaches, 2.5. the provision of objective assessment metrics, 2.6. enhancing proficiency in differential diagnosis abilities, 3. approaches for generating case-based mcqs, 3.1. generation through manual procedures.

• Selection of Topic: During the initial phase, educators execute a meticulous selection of a medical subject or issue that bears relevance to the curriculum, as indicated by Al-Rukban [ 7 ]. If the institution uses Learning Objectives, then these must also be noted to ensure that the questions are aligned with them.
• Development of Case Scenario: This phase entails the crafting of a patient scenario which could be derived from either authentic experiences or hypothetical situations, aiming to construct a contextual framework. Typical elements of a patient’s medical record integrate their medical history, vital statistics, laboratory results, and other pertinent information [ 14 ].
• Question Framing: The core objective of framing questions is to evaluate understanding, analysis, or application in connection with the presented case study [ 7 , 14 ].
• Distractor generation: Distractor conceptualization involves formulating conceivable incorrect alternatives (distractors) that are coherent and non-deceptive, a notion underscored by Al-Rukban and Kurdi [ 7 , 14 ].
• Validation: The refinement and validation of MCQs are optimized through a peer review, executed by educationalists and clinical experts. This collaborative methodology ascertains the enhancement of question clarity, accuracy, and pertinence [ 14 ].

3.2. Challenges of Manual Generation

3.3. the process of automated generation, 3.4. the advantages and obstacles associated with automated generation, 4. principles for designing case-based mcqs, 4.1. authenticity of cases, 4.2. precision and clarity in language, 4.3. pertinent and rigorous distractors, 4.4. incorporation of clinical reasoning, 4.5. facilitation of feedback, 4.6. conformity with educational objectives, 4.7. recurrent evaluation and validation, 5. validity and reliability of case-based mcqs, 5.1. validity of case-based mcqs.

• Content validity: This measures the extent to which the MCQs cover the topics and relevant clinical areas. It is important to select cases that show the many different situations that doctors might face [ 6 ].
• Criterion-related validity: This measures the extent to which the results from these MCQs match results from other tests measuring the same skills or knowledge. For example, you could check how well scores from a case-based MCQ test compare with scores from a hands-on clinical exam, following the ideas of Messick et al. [ 26 ]. Criterion-related validity can be further divided into concurrent validity (comparing to an established tool) and predictive validity (for predicting future outcomes) [ 27 ].
• Construct validity: This measures the extent to which the MCQs test the theoretical ideas that are supposed to be assessed. The questions should be able to effectively check if the intended theoretical concepts are understood by the students. For example, if a collection of MCQs are formulated with the intention of evaluating clinical reasoning abilities, it may be hypothesized that students who achieve higher scores on these assessments will possess superior clinical reasoning skills compared to those who obtain lower scores [ 28 ].

5.2. Reliability of Case-Based MCQs

• Test–retest reliability: Test–retest reliability refers to the assessment of the consistency of scores obtained by students when they take the same test on several occasions. According to Nunnally, a strong correlation between the MCQs indicates that they yield consistent outcomes throughout different time periods [ 29 ].
• Internal consistency: Internal consistency refers to the degree to which several items within an MCQ test produce consistent outcomes. Cronbach’s alpha is a frequently employed statistical measure for this particular objective. According to Nunnally, a high result, often over 0.7, signifies that the MCQs effectively assess the same underlying construct [ 29 ]. For test reliability, however, the Kuder–Richarson 20 (KR20) (when item difficulty is variable) and the KR21 (when the item difficulty is similar) are preferred [ 30 ].
• Inter-rater reliability: Inter-rater reliability is of utmost importance in situations where questions are open-ended and necessitate manual scoring. The measure assesses the level of consensus among several raters or examiners. According to Kurdi et al., a high coefficient of inter-rater reliability signifies a consistent scoring pattern among many examiners [ 6 ].

6. Challenges and Controversies in Case-Based MCQs

• Overemphasis on Recall: One potential issue with MCQs is the tendency to place excessive emphasis on recall. Although MCQs are effective in evaluating a wide range of content, there is a valid concern that they primarily measure memorization rather than comprehensive comprehension or practical application [ 3 ]. In order for case-based MCQs to be truly effective, it is imperative that they redirect their emphasis from mere memory to the domains of application and synthesis.
• The Importance of Misleading Distractors: The inclusion of effective distractors plays a vital role in enhancing the discriminatory capacity of MCQs. Nevertheless, it is worth noting that inadequately formulated distractors have the potential to deceive students, transforming questions into assessments of test-taking abilities rather than evaluating their clinical reasoning capabilities [ 31 ].
• Dependence on Stem Clarity: The importance of stem clarity in determining dependence is of utmost significance. The presence of ambiguous or unnecessarily lengthy stems in academic assessments may lead to unanticipated challenges, which could potentially put learners at a disadvantage [ 31 ].
• Cultural and Socio-Economic Biases: MCQs may unintentionally include cultural or socio-economic biases, thereby reflecting the perspectives of the individuals who authored the questions rather than universally accepted medical information. Kim and Zabelina (2015) argue that prejudices have the potential to create disadvantages for specific student groups [ 32 ].
• Over-Reliance on Single Best Answer: The practical situations encountered in the field of medicine rarely possess unequivocal resolutions. According to Scott et al. (2018), the utilization of single-best-answer MCQs may occasionally result in an oversimplification of intricate clinical settings [ 8 ].
• Security Concerns: Security concerns have emerged due to the widespread use of digital platforms and student collaboration tools, which have raised apprehensions over the security of exams and the potential for question-sharing. These concerns pose significant risks to the overall integrity of the assessment process [ 33 ].
• Technology Dependence: The integration of MCQs into digital platforms has led to a heightened reliance on technology. This presents difficulties pertaining to software malfunctions, user interface usability, and the issue of digital equity [ 34 ].

7. Future Directions

8. conclusions, author contributions, institutional review board statement, informed consent statement, data availability statement, conflicts of interest.

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Share and Cite

Al Shuriaqi, S.; Aal Abdulsalam, A.; Masters, K. Generation of Medical Case-Based Multiple-Choice Questions. Int. Med. Educ. 2024 , 3 , 12-22. https://doi.org/10.3390/ime3010002

Al Shuriaqi S, Aal Abdulsalam A, Masters K. Generation of Medical Case-Based Multiple-Choice Questions. International Medical Education . 2024; 3(1):12-22. https://doi.org/10.3390/ime3010002

Al Shuriaqi, Somaiya, Abdulrahman Aal Abdulsalam, and Ken Masters. 2024. "Generation of Medical Case-Based Multiple-Choice Questions" International Medical Education 3, no. 1: 12-22. https://doi.org/10.3390/ime3010002

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A Case Study Progression to Multiple Myeloma

Mary ann yancey.

Medical Oncology Branch at the Center for Cancer Research at the National Cancer Institute of the National Institutes of Health in Bethesda, MD.

Adam J. Waxman

Ola landgren.

Multiple myeloma consistently is preceded by precursor states, which often are diagnosed incidentally in the laboratory. This case report illustrates the clinical dilemma of progression from precursor to full malignancy. The article also discusses future directions in management and research focusing on myelomagenesis.

Multiple myeloma, characterized by the clonal proliferation of plasma cells in the bone marrow, is the second most common hematologic malignancy, with more than 20,000 new cases diagnosed per year in the United States ( Laubach, Richardson, & Anderson, 2010 ). Typical clinical features include anemia, renal failure, hypercalcemia, and skeletal lytic lesions ( Kyle & Rajkumar, 2004 ) (see Table 1 ). Despite significant advances in treatment, multiple myeloma has high morbidity and mortality reflected by an overall length of survival of about four to seven years ( Kumar et al., 2008 , Turesson, Velez, Kristinsson, & Landgren, 2009 ).

Diagnostic Criteria for Multiple Myeloma and Precursor States

MGUS—monoclonal gammopathy of undetermined significance; MM—multiple myeloma; SMM—smoldering multiple myeloma

Note . From “Criteria for the Classification of Monoclonal Gammopathies, Multiple Myeloma and Related Disorders” by the International Myeloma Working Group, 2003 , British Journal of Haematology , 121, pp. 752–753. Copyright 2003 by John Wiley and Sons. All rights reserved. Adapted with permission.

Multiple myeloma has two precursor states: monoclonal gammopathy of undetermined significance (MGUS) and smoldering multiple myeloma. MGUS is asymptomatic and affects about 3% of Caucasians older than age 50. MGUS has a 1% average annual risk for progression to multiple myeloma or related lymphoproliferative malignancies ( Kyle et al., 2002 , 2007 ) (see Figure 1 ). Two independent studies showed that multiple myeloma consistently is preceded by MGUS ( Landgren et al., 2009 ; Weiss, Abadie, Verma, Howard, & Kuehl, 2009 ). Smoldering multiple myeloma is another asymptomatic precursor to multiple myeloma with a substantially higher annual risk of progression. Smoldering multiple myeloma is defined by a monoclonal-protein concentration of 3 g/dl or higher or 10% or higher bone marrow plasma cells in the absence of end-organ damage ( International Myeloma Working Group, 2003 ). To date, an estimated 3,000 smoldering multiple myeloma cases are diagnosed annually in the United States; however, the numbers likely are not reliable because of prior inconsistent diagnostic criteria and under diagnosis from the malignancy’s asymptomatic nature ( Rajkumar, Lacy, & Kyle, 2007 ). Based on retrospective data from the Mayo Clinic, the average annual risk of progression from smoldering multiple myeloma to multiple myeloma is 10% for the first five years following smoldering multiple myeloma diagnosis, decreasing to 3% annually for the following five years, and becoming the same 1% annual rate of progression as MGUS thereafter ( Kyle et al., 2007 ).

Note . From “Clinical Course and Prognosis of Smoldering (Asymptomatic) Multiple Myeloma,” by R.A. Kyle, E.D. Remstein, T.M. Therneau, A. Dispenzieri, P.J. Kurtin, J.M. Hodnefield, and S.V. Rajkumar, 2007, New England Journal of Medicine, 356 , p. 2582. Copyright 2007 by Massachusetts Medical Society. All rights reserved.

In the late 1990s, Mr. A, an otherwise healthy 65-year-old Caucasian man, underwent a regular health check-up. As part of the blood work, serum protein electrophoresis was conducted. During the work-up, a monoclonal protein was detected and confirmed with immunofixation electrophoresis. The monoclonal protein was defined as immunoglobulin-G kappa with a concentration of 1.25 g/dl; the quantitative uninvolved immunoglobulin levels all were found to be normal. In addition, the complete blood count revealed normal hemoglobin, calcium, and creatinine levels. Mr. A was diagnosed with MGUS. Given the features of the serum protein abnormalities, Mr. A was recommended to have annual follow-up appointments to monitor his blood work. For several years, his monoclonal-protein level stayed in the range of 1–2 g/dl and he did not have other laboratory abnormalities or symptoms.

In early 2009, Mr. A’s monoclonal-protein level increased to 3.2 g/dl. He underwent a bone marrow biopsy with immunohistochemistry (CD138, kappa and lambda stains). Results of the biopsy showed 30%–40% plasma cells with kappa light-chain restriction. In addition, flow cytometry of the bone marrow aspirate showed that more than 99% of the plasma cells were abnormal. Serum calcium and creatinine levels were within normal limits. Albumin level was 3.2 g/dl, and the beta-2-microglobulin level was 2.3 mg/L. Skeletal survey was negative for lytic lesions. Taken together, the observations changed Mr. A’s diagnosis from MGUS to smoldering multiple myeloma.

After diagnosis, Mr. A was monitored every two months and assessed with serum protein electrophoresis, immunofixation electrophoresis, and routine laboratory tests (including calcium, albumin, complete blood count, and creatinine). The monoclonal-protein concentration increased by 0.3–0.5 g/dl at each visit, whereas calcium levels gradually increased and hemoglobin gradually decreased, despite the absence of clear symptoms.

In late 2009, Mr. A reported diffuse back pain and unspecific weak pain from the pelvis, so magnetic resonance imaging was done to exclude spinal cord compression; the result was negative. An 18-F-fluoro-deoxy-2-glucose positron-emission tomography/computed tomography (18-FDG PET/CT) scan had been done in early 2009 when the monoclonal-protein levels started to increase; the examination showed suspicious lesions in the vertebrae, whereas the skeletal survey was negative. Another 18-FDG PET/CT scan was conducted to compare to the previously noted abnormalities in the vertebrae. The scan revealed low intensity focal lesions in the sacrum and left iliac bone consistent with lytic lesions; again, the skeletal survey was negative for lytic lesions. According to diagnostic criteria, either lytic lesions or “abnormal compression fractures of vertebrae” detected by skeletal survey qualify for a diagnosis of multiple myeloma ( International Myeloma Working Group, 2003 ). In the context of Mr. A’s increasingly abnormal laboratory values, increased focal lesions on PET/CT, and symptoms in the area of abnormality, he was interpreted biologically and clinically as having multiple myeloma. He then began treatment for multiple myeloma.

Progression to Multiple Myeloma

To date, predicting whether an individual patient with MGUS or smoldering multiple myeloma will ultimately progress to multiple myeloma is not possible. However, two schemas have been developed to help define patients at higher or lower than average risk of progression (see Tables 2 and ​ and3). 3 ). Using the results of serum-protein assays including quantity of monoclonal protein, type of immunoglobulin heavy chain, and serum-free light chain ratio, as well as percentage of bone marrow plasma cells by biopsy in smoldering multiple myeloma, the Mayo Clinic developed a scheme to stratify patients based on risk of progression to multiple myeloma ( Dispenzieri et al., 2008 ; Rajkumar et al., 2005 ). The Spanish Programa Para el Estudio de la Terapéutica en Hemopatía Maligna (Program for the Study of Therapy in Hematologic Malignancies) (PETHEMA) study group developed a risk stratifications scheme based primarily on the results of immunophenotyping by flow cytometry, which requires a bone marrow aspirate sample ( Perez-Persona et al., 2007 ). Mr. A was at the highest risk for progression to multiple myeloma by the PETHEMA scheme (95% or higher abnormal plasma cells and decreased uninvolved immunoglobulins) and intermediate risk for progression by the Mayo Clinic criteria (monoclonal protein 3 g/dl or higher, bone marrow plasma cells 10% or higher, but serum-free light chain ratio lower than 8).

Monoclonal Gammopathy of Undetermined Significance to Multiple Myeloma: Predictors of Progression

PETHEMA—Programa Para el Estudio de la Terapéutica en Hemopatía Maligna (Program for the Study of Therapy in Hematologic Malignancies)

Note . Based on information from Perez-Persona et al., 2007 ; Rajkumar et al., 2005 .

Smoldering Multiple Myeloma to Multiple Myeloma: Predictors of Progression at Five Years

Note . Based on information from Dispenzieri et al., 2008 ; Perez-Persona et al., 2007 .

Although no formal guidelines exist for evaluation and follow-up, the Mayo Clinic recommends follow-up at two- to three-month intervals for the first year with monitoring of monoclonal-protein levels, serum chemistry, complete blood count, and urine protein studies ( Blade, Dimopoulos, Rosinol, Rajkumar, & Kyle, 2010 ). After that time, follow-up intervals may be extended to up to six months and maintained or reduced based on the patient’s risk profile. Because of the heterogeneous prognosis, thoughtful patient education is critical at all visits.

Outside of clinical trials, patients with smoldering multiple myeloma should not be treated unless progression to multiple myeloma occurs ( Kyle et al., 2007 ). Current guidelines do not endorse treatment of patients with smoldering multiple myeloma ( Kyle et al., 2007 ). In fact, prior clinical trials have not shown an increase in overall survival for patients with smoldering multiple myeloma treated with melphalan-prednisone, thalidomide, or bisphosonates, but zolendronate therapy decreased the risk of pathologic fractures following progression ( Barlogie et al., 2008 ; Hjorth et al., 1993 ; Musto et al., 2008 ). Preliminary data from an ongoing phase III trial using lenalidomide-dexamethasone versus observation in high-risk smoldering multiple myeloma has shown lower risk of progression for patients assigned to the treatment arm; overall survival data are not yet available ( Mateos et al., 2009 ). The goal of treating smoldering multiple myeloma is to cure the disease outright or to manage it chronically, delaying progression to full myeloma and improving survival. However, clinicians should consider the possibilities of significant drug toxicities, development of refractory disease, and long-term adverse events in asymptomatic patients ( Waxman, Kuehl, Balakumaran, Weiss, & Landgren , in press).

Multiple myeloma is a diagnosis based on clinical, laboratory, and radiographic assessment for myeloma-related end-organ damage. In contrast, MGUS and smoldering multiple myeloma often are incidental laboratory diagnoses based on presence and size of the monoclonal protein and percentage of bone marrow plasma cells. Current criteria for the diagnosis of MGUS, smoldering multiple myeloma, and multiple myeloma were developed by the International Myeloma Working Group in 2003. However, as in the case of Mr. A, the clinical picture can be highly complex, requiring very careful history and physical examination correlated with laboratory values and newer imaging studies.

Prospective molecular profiling studies are ongoing, with the expectation of gaining more accurate risk stratification for individuals with MGUS and smoldering multiple myeloma. Knowledge of present and future predictors of risk for progression may allow for better monitoring of patients with myeloma precursor disease. Future goals are to identify high-risk individuals and develop effective therapies with limited side effects aimed at delaying progression or cure in high-risk patients. At the National Institutes of Health in Bethesda, MD, ongoing studies focus on molecular profiling and the development of novel therapies for high-risk myeloma precursor disease. For more information, visit www.clinicaltrials.gov .

Acknowledgments

The authors take full responsibility for the content of the article. This work was supported by the Intramural Research Program of the National Cancer Institute of the National Institutes of Health. The content of this article has been reviewed to ensure that it is balanced, objective, and free from commercial bias. No financial relationships relevant to the content of this article have been disclosed by the editorial staff.

Contributor Information

Mary Ann Yancey, Medical Oncology Branch at the Center for Cancer Research at the National Cancer Institute of the National Institutes of Health in Bethesda, MD.

Adam J. Waxman, Medical Oncology Branch at the Center for Cancer Research at the National Cancer Institute of the National Institutes of Health in Bethesda, MD.

Ola Landgren, Medical Oncology Branch at the Center for Cancer Research at the National Cancer Institute of the National Institutes of Health in Bethesda, MD.

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Case report

• Open access
• Published: 23 March 2023

Vitamin E-induced coagulopathy in a young patient: a case report

• Ritika Abrol 1 ,
• Reshma Kaushik   ORCID: orcid.org/0000-0001-7826-2092 1 ,
• Deepak Goel 2 ,
• Sonu Sama 3 ,
• Rajeev Mohan Kaushik 1 &
• Mansi Kala 4  

Journal of Medical Case Reports volume  17 , Article number:  107 ( 2023 ) Cite this article

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High-dose vitamin E intake is known to inhibit vitamin K-derived coagulation factor synthesis, which can cause serious bleeding events such as gastrointestinal bleeding and intracranial hemorrhage. We report a case of coagulopathy induced by marginally increased levels of vitamin E.

Case presentation

A 31-year-old Indian man presented with oral bleeding, black tarry stools, and bruising over his back. He had been taking non-steroidal anti-inflammatory drugs for low backache and vitamin E for hair loss. He had mild anemia with normal platelet count, thrombin time, and prolonged bleeding time, activated partial thromboplastin time, and prothrombin time. Serum fibrinogen was slightly raised. Mixing studies with pooled normal plasma, aged plasma, and adsorbed plasma were suggestive of deficiency of multiple coagulation factors due to acquired vitamin K deficiency. Serum phylloquinone was normal, while prothrombin induced by vitamin K absence-II level was increased. Serum alpha-tocopherol was slightly raised. Upper gastrointestinal endoscopy showed multiple gastroduodenal erosions. A final diagnosis of vitamin E toxicity-related coagulopathy was made. The patient responded well to pantoprazole, vitamin K supplementation, multiple fresh frozen plasma transfusions, and other supportive treatments besides the discontinuation of vitamin E supplementation. The coagulation parameters normalized, and the patient was discharged with complete resolution of symptoms and remained asymptomatic during the follow-up for 6 months.

Conclusions

Vitamin E-related inhibition of vitamin K-dependent factors with coagulopathy may occur even at marginally increased levels of serum vitamin E. This risk becomes significant in patients receiving other drugs that may increase the risk of bleeding.

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Introduction

Vitamin E toxicity is rarely observed due to its ready excretion in bile and urine [ 1 ]. However, it has been noted to occur in cases of lipid or hepatobiliary disorders where its absorption or excretion may be altered. High-dose vitamin E supplementation is known to inhibit vitamin K-related enzyme activation relevant for coagulation factor syntheses, such as factors II, VII, IX, and X, as well as protein C and S. Deficiency of factors II, VII, IX, and X promotes bleeding [ 2 ], while deficiency of protein C and protein S leads to the loss of their natural anticoagulant effects, with consequent unrestrained thrombin generation, resulting in thromboembolism [ 3 , 4 ]. Generally, deficiency of factors II, VII, IX, and X is the dominant effect and may lead to bleeding. The mechanism of inhibition of vitamin K-related enzyme activation relevant for coagulation factor synthesis is evidenced by the elevation of levels of prothrombin induced by vitamin K absence-II (PIVKA-II), due to an increase in under-gamma-carboxylated prothrombin.

Vitamin E excess further inhibits factor IX activation by reducing vitamin K-dependent carboxylation of glutamate and inhibits platelet aggregation [ 5 ]. Thus vitamin E toxicity can present as coagulopathies alongside general complaints such as malaise, nausea, myalgia, and fatigue [ 6 ].

Though it is generally understood that vitamin E toxicity may occur if consumed at levels greater than 1000 mg/day, there is no fixed cut-off point [ 7 ]. The circulating alpha-tocopherol levels depend heavily on the lipid content of the blood. Vitamin E cannot be measured accurately by circulating alpha-tocopherol levels in patients who have very high or very low cholesterol levels. The same applies to patients with average cholesterol levels as well. This is because of the upregulation of biliary and urinary excretion once vitamin E levels increase in the body [ 5 ]. Because of these irregularities in vitamin E metabolism, the minimum dose, form, and duration of vitamin E intake required to induce a clinically significant effect on coagulation pathways are not known [ 7 ].

Vitamin E toxicity may be overlooked as the cause of coagulopathy if serum levels of vitamin E are only marginally increased. We report a case of coagulopathy induced by marginally increased levels of serum vitamin E.

A 31-year-old Indian man, a restaurateur by profession, presented to the emergency services with complaints of low backache for 1 month and oral bleeding, the passage of black tarry stools, and bruising over his back for the last seven days. He had intense nausea and fatigue for several days and began to develop bruising about 1 week ago. He experienced oral bleeding and passed black tarry stools three times, following which he decided to visit the emergency services.

The backache was non-specific in nature. The patient had been taking over-the-counter non-steroidal anti-inflammatory drugs (NSAIDs) in the form of ibuprofen 400 mg infrequently for the past 1 month for his backache, before his current symptoms of bleeding.

The patient had no complaints of bleeding tendencies in the past, such as frequent nosebleeds, prolonged bleeding from gums or injuries, gastrointestinal bleeds, bleeding into joints, easy bruising, and hematuria. There was no history of jaundice, peptic ulcer, malignancy, or bony aches and pains. He had never received any blood transfusions. There was no history of the use of anticoagulants, antiplatelet drugs, and corticosteroids. There was no history of heavy exposure to pesticides, injury, or surgery.

The patient also reported the use of vitamin E supplementation for his complaints of recent onset hair loss and dryness of the skin. He had taken vitamin E 400 mg twice daily for a period of 2 months, which was accompanied by a fairly poor diet due to work-related stressors.

He was a non-smoker. He drank alcohol sparingly and had no drug addictions. He consumed a mixed diet. He was residing in a semiurban area that had moderate air pollution levels.

He was a bachelor and had two elder brothers. His parents and brothers were healthy. There was no family history of any bleeding disorder in either parents, maternal grandparents, uncles, aunts, or siblings. There was no history of any consanguineous marriage in the family.

On admission, the patient was hemodynamically stable. He was well-built and in no distress. His pulse rate was 74 beats per minute, blood pressure was 122/78 mmHg, and oral temperature was 37 °C. He had nonspecific thinning of hair over bitemporal regions. Mild pallor and bleeding from the gums were present. Multiple punctate, perifollicular hemorrhages were present over his forearms (Fig.  1 ) along with large ecchymotic patches over his back (Fig.  2 ). Icterus, cyanosis, clubbing, lymphadenopathy, telangiectasis, signs of chronic liver disease, and pedal edema were absent. Neurological examination showed normal higher mental functions with a mini-mental state examination (MMSE) score of 28/30. The flapping tremor was absent. The cranial nerve examination was normal. No ocular movement dysfunction or nystagmus was seen. There were no fasciculations, motor deficits, or incoordination. Tendon jerks were normal and the plantars showed bilateral flexor response. Sensations were intact.

Petechiae over the arm

Ecchymosis over the back of the patient

Examination of the cardiovascular system and the respiratory system was normal. Abdominal examination showed no hepatomegaly, splenomegaly, or ascites. He was evaluated for gingival bleeding and melena.

The laboratory work-up showed low hemoglobin (11.0 g/dL) with normal mean corpuscular volume (87.2 fl), mean corpuscular hemoglobin (28.9 pg), leukocyte count (9900/mm 3 ), and platelet count (2,72,000/mm 3 ). Random blood sugar, liver and renal function tests, and thyroid profile were within normal range. The anemia was evaluated further. Peripheral blood film showed normocytic normochromic red blood cells, with normal white blood cells and platelets. Serum iron was 39 µg/dL, ferritin 22 µg/L, total iron binding capacity 305 µg/dL, and lactate dehydrogenase 277 U/L. Anemia was attributed to his bleeding. Bleeding time (BT) was prolonged (7 minutes, reference range 2–6 minutes). Also activated partial thromboplastin time (APTT) and prothrombin time (PT) were significantly prolonged (APTT 104.5 seconds, reference range 29–33 seconds; PT > 1 minute, reference range 11.4–14.8 seconds). Thrombin time (TT) was normal (18 seconds, reference range 14–20 seconds). Mixing studies were performed with pooled normal plasma, aged plasma, and adsorbed plasma. Full correction for APTT and PT occurred with pooled normal plasma. Mixing studies with aged plasma as well as adsorbed plasma did not show correction of abnormal PT of the patient’s plasma and were suggestive of deficiency of multiple factors. Factor VIII assay was 100% (reference range 50–150%). Factor IX assay was 9% (reference range 50–150%). The serum fibrinogen level was slightly raised (430 mg/dL, reference range 200–400 mg/dL). Due to the unavailability of resources, further factor assays were not performed.

A possibility of acquired vitamin K deficiency was considered in view of the deficiency of multiple coagulation factors. The patient was investigated further to elucidate the etiology. Serum phylloquinone (vitamin K1) level was normal (1.14 ng/mL, reference range 0.10–2.20 ng/mL). Serum PIVKA-II level was measured by chemiluminescent microparticle immunoassay (CMIA) and was increased (326.7 mAU/mL, upper normal limit 40 mAU/mL). Serum alpha-tocopherol, which is considered a surrogate marker for serum vitamin E level, was evaluated and found to be slightly raised (20.10 mg/L, reference range 5–18 mg/L).

Ultrasound examination of the abdomen was normal. Upper gastrointestinal endoscopy showed few gastroduodenal erosions with no active bleeding. Urinalysis did not show any abnormality. The stool was negative for occult blood.

Chest X-Ray and X-Rays of the lumbosacral spine were normal. The patient was further evaluated for ankylosing spondylitis, in view of the history of backache, to identify the association of any collagen-vascular disease with his bleeding complaint, and was found to be negative for HLA-B27.

Given the patient’s history, presenting complaints, and investigation findings, a final diagnosis of vitamin E toxicity-related coagulopathy was made. Exogenous vitamin E supplementation was stopped. The patient received intravenous fluids, pantoprazole, vitamin K, vitamin C, multiple transfusions of fresh frozen plasma (FFP), and other supportive treatments.

The patient’s complaints were alleviated with the treatment and the coagulation parameters normalized. The patient was discharged with complete resolution of symptoms and remained asymptomatic during the follow-up for 6 months.

This male patient presented with oral bleeding, melena, and bruising over his back. He was taking NSAIDs infrequently for low backache, and vitamin E for hair loss. Investigations suggested the deficiency of multiple coagulation factors due to acquired vitamin K deficiency. Upper gastrointestinal endoscopy showed gastroduodenal erosions. On the basis of normal serum phylloquinone, increased PIVKA-II levels, and slightly raised serum alpha-tocopherol, the patient was diagnosed as having vitamin E toxicity-related coagulopathy, which responded well to pantoprazole, vitamin K supplementation, fresh frozen plasma transfusions, and other supportive treatment besides the discontinuation of exogenous vitamin E administration. As such, this case documents the inhibition of vitamin K-dependent factors with coagulopathy at only marginally increased levels of serum vitamin E, while vitamin E toxicity-related coagulopathy has been reported earlier at much higher levels of serum vitamin E [ 5 , 8 ].

The extent of vitamin E excess-induced vitamin K suppression varies as per the dose consumed. Any preexisting subclinical deficiency may be unmasked by any level of vitamin E excess, thereby implying that there may be considerable interpersonal variability reflecting the inhibitory effects of vitamin E on individuals with a poor vitamin K status [ 9 ].

Though serum alpha-tocopherol level was only marginally increased and did not confirm the vitamin E-related toxicity convincingly, it is possible that the actual level of vitamin E may be higher than measured, as alpha-tocopherol may not measure the circulating vitamin E accurately.

Vitamin E may compete for the enzyme vitamin K epoxide reductase, which converts the precursors of vitamin K into the active form of vitamin K [ 3 ]. As a result, the activation of inactive vitamin K may be inhibited, thereby preventing the gamma-carboxylation, and thus activation, of vitamin K-dependent coagulation factors. The inability to activate the clotting cascade via these factors may increase the risk of bleeding.

Moreover, vitamin E excess decreases the availability of factor IX as it reduces the production of glutamate, which is required for factor IX production [ 3 ]. Platelet aggregation is also decreased in patients receiving vitamin E and may be in part due to a mechanism linked to protein kinase C inhibition [ 10 ]. The bleeding risk due to a marginally elevated level of vitamin E was also aggravated by NSAIDs in our patient.

Normal serum phylloquinone and increased PIVKA-II levels negate the possibility that this patient had vitamin K deficiency leading to gastrointestinal bleeding/skin changes. Moreover, our subject was a young adult and there was no evidence of any of the causes of vitamin K deficiency in adults, such as malnutrition, lipid malabsorption, malignancy, or renal disease [ 11 ].

Prolongation of both PT and APTT, and a normal TT in our patient were suggestive of coagulation factor deficiency in the common pathway (factors II, V, VII, and X), along with factor IX deficiency or presence of inhibitors of prothrombin, fibrinogen, factor V, or factor X. Full correction of the abnormal APTT and PT by pooled normal plasma ruled out factor inhibitors and suggested a deficiency of coagulation factors. The absence of correction of abnormal PT on mixing aged plasma with the patient’s plasma suggested factor V deficiency. The absence of correction of PT on mixing adsorbed plasma with the patient’s plasma was suggestive of factor II, VII, and X deficiency. Moreover, selected assays showed normal factor VIII and raised factor I (fibrinogen) levels, but deficient factor IX levels.

Inherited factor deficiency was not likely because of the absence of any abnormal bleeding until the current episode. Acquired factor deficiency due to liver disease was unlikely in view of normal liver function tests and a normal ultrasound scan of the liver. The absence of thrombocytopenia and a raised serum fibrinogen level excluded the possibility of disseminated intravascular coagulation causing factor deficiency. A raised fibrinogen level may be due to low serum iron concentrations, caused by blood loss, as a negative correlation was observed between total fibrinogen and serum iron concentrations in a large-scale epidemiological study [ 12 ].

Anticoagulant-induced factor deficiency was ruled out as the patient was not receiving any anticoagulants. The presence of combined factor deficiencies generally implies severe vitamin K deficiency, but it would be reasonable to assume that a secondary factor must exist, as an isolated vitamin K deficiency is rare due to its continual production by the gut microbiome in healthy individuals [ 13 ].

Vitamin E excess potentiated by ill-timed NSAID use could have led to the precipitation of this patient’s complaints [ 14 ]. Vitamin E excess could have inhibited vitamin K-related coagulation factor synthesis and prolonged the BT, while NSAIDs could cause gastroduodenal erosions, besides prolonging the BT, and thus add to the risk of gastrointestinal bleeding.

Vitamin E toxicity-induced coagulopathy has been described at high levels of serum alpha-tocopherol, but there is a case report of intracranial hemorrhage attributed to vitamin E toxicity occurring at serum alpha-tocopherol level of 23.3 mg/L [ 5 ]. Bleeding manifestations occurred in our case at an even lower level of serum alpha-tocopherol. Our case report highlights the often underscored bleeding risks with vitamin E consumption and that this coagulopathy can also occur at marginally increased levels of vitamin E.

Vitamin E has been lauded for its various dermatological and cardiovascular benefits, all of which are exceptional [ 15 ]. However, the same stellar reputation leads to indiscriminate use by prescription, as well as without prescription, by patients. Though the bleeding risk may be hemodynamically relevant in patients already receiving anticoagulants, our case report reveals that the risk of upper gastrointestinal bleeding could also be increased in patients taking both NSAIDs and vitamin E. Moreover, bleeding from gums, ecchymotic patches over the back, normal serum phylloquinone level, and deficiency of multiple coagulation factors, in the presence of increased PIVKA-II level, were highly suggestive of coagulopathy due to marginally increased vitamin E-related inhibition of vitamin K-dependent factors.

As the patient recovered readily with vitamin K supplementation and FFP transfusions, a final diagnosis of vitamin E toxicity-related coagulopathy was reaffirmed, although NSAID intake might have been an aggravating factor.

Vitamin E-related inhibition of vitamin K-dependent factors with coagulopathy may occur even at slightly raised levels of vitamin E. This risk is augmented in presence of other drugs that have the potential to cause bleeding.

Thus, patients must be discouraged from taking medications without proper medical consultation. Physicians too must be wary of the potential interactions of substances generally considered harmless.

Availability of data and materials

Not applicable.

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Acknowledgements

We are grateful to the patient for giving his consent to publish this case. We thank Dr. Sadhana Raturi and Dr. Madhurima Kaushik for their valuable suggestions during the preparation of the manuscript.

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Department of General Medicine, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Dehradun, Uttarakhand, 248016, India

Ritika Abrol, Reshma Kaushik & Rajeev Mohan Kaushik

Department of Neurology, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Dehradun, Uttarakhand, 248016, India

Deepak Goel

Department of Critical Care Medicine, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Dehradun, Uttarakhand, 248016, India

Department of Pathology, Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Dehradun, Uttarakhand, 248016, India

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RK, SS, and RMK conceived the work. RK, SS, RA, DG, and MK contributed to the diagnosis and treatment. RA and SS prepared the initial manuscript. RK, RMK, and DG revised the manuscript critically for important intellectual content. All authors read and approved the final manuscript.

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Correspondence to Reshma Kaushik .

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Abrol, R., Kaushik, R., Goel, D. et al. Vitamin E-induced coagulopathy in a young patient: a case report. J Med Case Reports 17 , 107 (2023). https://doi.org/10.1186/s13256-023-03827-y

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DOI : https://doi.org/10.1186/s13256-023-03827-y

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• Coagulopathy
• Vitamin E toxicity

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• Inpatient case characteristics of SGLT2 inhibitor-associated diabetic ketoacidosis: a retrospective study
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• http://orcid.org/0009-0008-4991-7190 Zhongpei Yang 1 ,
• Weixia Zhang 2 ,
• Hefeng Chen 2 ,
• Qianwen Peng 2
• 1 Department of Pharmacy , The People's Hospital of QianNan , Duyun , China
• 2 Department of Pharmacy , Shanghai Jiao Tong University Medical School Affiliated Ruijin Hospital , Shanghai , China
• Correspondence to Dr Weixia Zhang, Department of Pharmacy, Shanghai Jiao Tong University Medical School Affiliated Ruijin Hospital, Shanghai, China; wxzhang2001{at}163.com

Objectives Diabetic ketoacidosis (DKA) is a serious complication in patients treated with sodium-glucose co-transporter 2 inhibitors (SGLT2i). The aim of this study was to investigate the relationship between SGLT2i and the risk of DKA, and to identify high-risk groups and characteristics that should be emphasised.

Methods A retrospective case series study was conducted to collect medical records of inpatients diagnosed with DKA and using SGLT2i before the onset of the disease from September 2022 to September 2023 in a tertiary hospital in Shanghai. Cases that met the inclusion criteria were retrieved through the electronic medical record system. Information was collected to compare the risk of DKA in patients with different characteristics.

Results A total of 21 patients (12 men and 9 women) met the criteria for SGLT2i-associated DKA. The mean diabetes duration was 10.4 years, with 47.6% (10/21) of patients diagnosed with euglycaemic DKA. The drug treatment regimen most commonly used was the combination of SGLT2i and metformin, representing 52.4% (11/21) of cases. The most common clinical symptoms were nausea, vomiting, abdominal pain and malaise. Common predisposing factors were acute infections, acute pancreatitis (predominantly hyperlipidaemic type), dietary inappropriateness, acute cardiovascular and cerebrovascular events and surgery. 71.4% of patients (15/21) had multiple risk factors.

Conclusion The use of SGLT2i in diabetic patients is associated with an increased risk of DKA, particularly in the presence of predisposing factors such as infection. Furthermore, long diabetes duration, decreased pancreatic β-cell function and the combined use of metformin may also contribute to the risk of DKA in patients treated with SGLT2i. The findings of this study provide valuable insights for better identification and management of DKA risks associated with SGLT2i in clinical practice.

• Diabetes Mellitus
• DRUG-RELATED SIDE EFFECTS AND ADVERSE REACTIONS
• HEALTH SERVICES ADMINISTRATION
• Pharmacovigilance
• Substance-Related Disorders

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https://doi.org/10.1136/ejhpharm-2024-004124

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Metrics details

None of the early M-Health applications are designed for case management care services. This study aims to describe the process of developing a M-health component for the case management model in breast cancer transitional care and to highlight methods for solving the common obstacles faced during the application of M-health nursing service.

We followed a four-step process: (a) Forming a cross-functional interdisciplinary development team containing two sub-teams, one for content development and the other for software development. (b) Applying self-management theory as the theoretical framework to develop the M-health application, using contextual analysis to gain a comprehensive understanding of the case management needs of oncology nursing specialists and the supportive care needs of out-of-hospital breast cancer patients. We validated the preliminary concepts of the framework and functionality of the M-health application through multiple interdisciplinary team discussions. (c) Adopting a multi-stage optimization strategy consisting of three progressive stages: screening, refining, and confirmation to develop and continually improve the WeChat mini-programs. (d) Following the user-centered principle throughout the development process and involving oncology nursing specialists and breast cancer patients at every stage.

Through a continuous, iterative development process and rigorous testing, we have developed patient-end and nurse-end program for breast cancer case management. The patient-end program contains four functional modules: “Information”, “Interaction”, “Management”, and “My”, while the nurse-end program includes three functional modules: “Consultation”, “Management”, and “My”. The patient-end program scored 78.75 on the System Usability Scale and showed a 100% task passing rate, indicating that the programs were easy to use.

Conclusions

Based on the contextual analysis, multi-stage optimization strategy, and interdisciplinary team work, a WeChat mini-program has been developed tailored to the requirements of the nurses and patients. This approach leverages the expertise of professionals from multiple disciplines to create effective and evidence-based solutions that can improve patient outcomes and quality of care.

Peer Review reports

Female breast cancer is the second leading cause of global cancer incidence in 2022, with an estimated 2.3 million new cases, representing 11.6% of all cancer cases [ 1 ]. Due to surgical trauma, side effects of drugs, fear of the recurrence or metastasis of breast cancer, changes in female characteristics, and lack of knowledge, patients with breast cancer frequently experience a series of physical and psychological health problems [ 2 , 3 , 4 , 5 , 6 ]. These health problems seriously affected patients’ life and work [ 7 , 8 ]. At present, community nursing in China is still in the developing stage, and the oncology specialty nursing service capacity of community nurses is not enough to deal with the health problems of breast cancer patients. It made continuous care for out-of-hospital breast cancer patients a weak link in the Chinese oncology nursing service system.

Nowadays, case management is employed to manage health problems for out-of-hospital breast cancer patients worldwide [ 9 , 10 , 11 , 12 , 13 , 14 , 15 ]. Case management involves regular telephone follow-ups and home visits by case management nurses to provide educational support to patients, thereby ensuring uninterrupted continuity of care [ 16 , 17 ]. The home visits and organization of patient information required for case management tasks consume a significant amount of time, manpower, and material resources [ 17 ]. In China, case management services are primarily undertaken by oncology nursing specialists from tertiary hospitals in their spare time [ 18 ]. However, the shortage of nurses has consistently been one of the major challenges facing the nursing industry in China, especially in tertiary hospitals [ 19 ]. Consequently, the implementation and promotion of case management in China also face great difficulties in reality [ 20 ].

The Global Observatory for eHealth (GOe) of the World Health Organization (WHO) defines mobile health (M-Health) as “medical and public health practice supported by mobile devices, such as mobile phones, patient monitoring devices, personal digital assistants (PDAs), and other wireless devices” [ 21 , 22 ]. With the development of digital technology and the COVID-19 pandemic in 2019, M-Health applications were further integrated into healthcare services, which increased the demand for M-Health applications in turn [ 23 , 24 ]. Compared with the traditional health service model, M-Health service model has the advantages of high-level informatization, fast response speed, freedom from time and location constraints, and resource-saving, etc. In the context of limited nursing human resources, M-Health service provides a new solution for the case management of out-of-hospital breast cancer patients [ 23 , 25 , 26 ].

Researchers have developed a range of M-Health applications targeting breast cancer patients. To our knowledge, none of these developed M-Health applications are designed for case management nursing services.

Early M-Health applications were mostly designed for single interventional goals, such as health education, medication compliance, self-monitoring, etc. Larsen et al. applied a M-Health application to monitor and adjust the dosage of oral chemotherapy drugs in breast cancer patients, and the results suggested that the treatment adherence was effectively improved [ 27 ]. Heo and his team successfully promoted self-breast-examination behavior in women under 30 years old using a M-Health application [ 28 ]. Mccarrol carried out a M-Health diet and exercise intervention in overweight breast cancer patients and found that the weight, BMI, and waist circumference of the intervention group decreased after one month [ 29 ]. Smith’s team found that their application promoted the adoption of healthy diet and exercise behaviors among breast cancer patients [ 30 ]. The application designed by Eden et al. enhanced the ability of breast cancer patients receiving chemotherapy to recognize adverse drug reactions [ 31 ]. Keohane and colleagues designed a health educational application based on the best practices and it proved effective in improving breast cancer-related knowledge [ 32 ]. The guideline-based M-Health application developed by Eden et al. optimized breast cancer patients’ individualized health decision-making regarding mammography [ 33 ].

With the progress of computer technology and the emphasis on physical and mental rehabilitation of breast cancer patients, some universities [ 34 , 35 ] in China have separately developed M-Health applications for comprehensive health management, which provide access to online communication, health education, and expert consultation.

Analyzing these developed applications deeply, three factors could be found that hindered the promotion of applications in real life. Firstly, the developing procedure usually lacks contextual analysis based on the actual usage context during the design phase. Secondly, there is a lack of consistent and long-term monitoring and operation staff in the subsequent program implementation. These factors may be the main reasons why many M-Health applications face difficulties in promotion and continuous operation after the research phase. Furthermore, as applications need to be installed on patients’ smartphones, certain hardware requirements, such as memory, may also pose restrict the adoption of M-Health applications to some extent.

In order to meet the needs of supportive care for out-of-hospital breast cancer patients and the needs of case management for oncology nurse specialists, we formed a multidisciplinary research team and collaboratively developed a WeChat mini-program for breast cancer case management in the CMBM (M-health for case management model in breast cancer transitional care) project. WeChat is chosen as the program development platform based on the following considerations. Firstly, WeChat is the most popular and widely used social software in China. As of December 31, 2020, the monthly active users of WeChat have exceeded 1.2 billion, and the daily active users of WeChat mini-programs exceeded 450 million [ 36 ]. Secondly, users can access and use the services of the mini-program directly within the WeChat platform, without the need to download or install additional mobile applications. This reduces the hardware requirements for software applications. The above two factors allow for a positive user experience and a realistic foundation for software promotion.

The purpose of this study is to describe the process of developing a tailored M-health component for the case management model in breast cancer transitional care and to highlight methods for solving the common obstacles faced during the application of M-health nursing service.

Methods and results

The development process was conducted in four steps: (a) An interdisciplinary development team was formed, consisting of two sub-teams dedicated to content and software development. (b) Using the self-management theory as the theoretical framework, contextual analysis was used to understand the case management needs of oncology nursing specialists and the supportive care needs of out-of-hospital breast cancer patients. Through iterative discussion within the interdisciplinary team, the preliminary conception of the application framework and function was formed. (c) A multi-stage optimization strategy was adopted to develop and regularly update the WeChat mini-programs, including three stages (screening, refining, and confirming). (d) During the entire development process, a user-centered principle was followed with the involvement of oncology nursing specialists and breast cancer patients, including development, testing, and iterative development phases.

The interdisciplinary team

An important prerequisite for developing M-health applications is the formation of an interdisciplinary development team. We built a multidisciplinary team consisting of researchers, oncology nursing specialists, and software developers. Each team member brought their expertise from their respective fields, and all individuals were considered members of the same team rather than separate participants with a common goal.

Two sub-teams were established, one responsible for content development, and the other for software development. The content development team consisted of researchers and six senior breast oncology nursing specialists with bachelor’s degrees and over 10 years of clinical experience. Their work included contextual analysis, functional framework design, and content review of the “Information” module. The software development team included researchers and experienced software developers. Their tasks involved developing the mini-program based on the functional framework and requirements designed by the content development team.

The development team used contextual analysis to identify the actual usage needs of two target groups for the mini-program: oncologist nurse specialists and out-of-hospital breast cancer patients.

Involvement of oncology nursing specialists and breast cancer patients following user-centered design principle

Since the oncology nursing specialists and breast cancer patients are targeted users of the mini-program, the two groups fully participated in the development according to the user-centered principle. Nursing specialists who in charge of case management were interviewed about the preliminary functional framework of the mini-program. The interview results are presented in the section “Driving the Development Process via the Contextual Analysis Findings.” Semi-structured in-depth interviews were conducted in the testing and iteration stage to gain user feedback from nursing specialists to improve the applicability and usability of the mini-program. The interview guide can be found in the supplementary material.

Breast cancer patients fully engaged in the three developing phases (Screening, Refining, and Confirming). In the Screening Phase, since the self-management theory was selected as the theoretical framework, the supportive care needs of out-of-hospital breast cancer patients were explored, and the functional framework of the mini-program was constructed accordingly. In the Refining Phase, patients were invited to evaluate the usability and practicality of the mini-program through system tests and semi-structured in-depth interviews. The results of the system test are presented in the Results of System Test section. The feedback from interviews and corresponding iterative updates are listed in Table  1 . In the Confirming Phase, our research team is conducting clinical trials in out-of-hospital breast cancer patients to find out the actual effect of the mini-program on recovery.

The theory framework of the mini-program

This study applied the self-management theory [ 37 ] as the theoretical framework. The self-management theory explains how individual factors and environmental factors influence an individual’s self-efficacy, which ultimately affects the generation and development of individual behaviors. Self-efficacy is influenced by direct experience, indirect learning, verbal persuasion, and psychological arousal. By providing individuals with sufficient knowledge, healthy beliefs, skills, and support, their self-efficacy is increased, and they are likely to engage in beneficial health behaviors and self-management. Individuals who are confident in their abilities to apply self-management behaviors and overcome obstacles by improving their self-management skills and persevere in their efforts to manage their health [ 37 ]. Self-efficacy is directly and linearly positively related to the active adoption of health management behaviors [ 38 ]. The functions of the various parts of the mini-program designed using self-management theory can broaden the pathways and levels of efficacy information generation in four ways: direct experience, indirect learning, verbal persuasion, and mental arousal. Patients with high self-efficacy will take positive steps to achieve desired goals and possess disease-adapted behaviors. The form of the mini-application function block diagram is shown in Fig.  1 .

Driving the development process via the contextual analysis findings

Contextual analysis [ 39 ] is a method of discerning the profound significance and influence of language, behavior, events, and so forth, by examining them within a particular environment or background. Rather than being an afterthought, contextual analysis sheds light on the meaning and inner dynamics of our primary subject of interest. Through contextual analysis, we can gain a deeper understanding of the user’s usage scenarios, including their motivations, goals, environment, and behavior. This helps us better understand user needs, as well as the problems and challenges they may encounter when using the software.

In this paper, we adopted contextual analysis to gain a detailed understanding of the needs of oncology nurse specialists and out-of-hospital breast cancer patients. The research team adopted a mixed research strategy to achieve contextual analysis of the target users. A cross-sectional study was conducted among 286 patients and qualitative semi-structured in-depth interviews were applied in 12 patients to find out the supportive care needs of out-of-hospital breast cancer patients. According to the contextual analysis results from patients, the functional framework of the mini-program was constructed. See Fig.  2 for details.

Supportive care needs of out-of-hospital breast cancer patients

Contextual analysis of breast cancer case management nurses was conducted through focus group interview. The interview results were listed as three themes: health information, personal self-management, and case management needs. Health information included breast cancer-related knowledge, the side effects of chemotherapy drugs, and symptom management measures. The key task of personal self-management contained temperature monitoring, weight management, functional exercise, and symptom management. Case management needs involved storage and management of patients’ medical records and development of a nurse-end program.

Based on the contextual analysis results of out-of-hospital breast cancer patients and the oncology case management nurses, the framework and functional block of the mini-program were formed. An overview of the CMBM Software development process is listed in Fig.  3 .

Overview of the CMBM software development process

Patient-end program functional modules

Using the results of the contextual analysis, we design the functional modules of the patient-end program based on the patient’s supportive care needs. For example, the “Information” section is designed to meet the “Information need” of breast cancer patients; the “social needs” and “spiritual needs” of patients suggest that breast cancer patients lack peer support, and for this reason, the"Interaction” section for patients has been added to the app to provide a communication platform for patients.

The patient-end program include four functional modules: “Information”, “Interaction”, “Management” and “My”. In the “Information” module, information about breast cancer treatment and health management are compiled based on clinical guildlines. The “Interaction” module allows patients to interact with fellow patients and consult an case management nurse. In the “Management” module, patients can record and review their self-management-related health status, including three medical parameters (temperature, blood pressure, weight) and three behavioral parameters (daily steps, medication, mindfulness excersice). The “My” module enables patients to input and edit their basic personal information and medical history. The main structure and information support module contents are listed in Fig.  4 .

The main menu of patient-end program

Nurse-end program functional modules

The design of the functional modules of the nurse-end program was also derived from the results of contextual analyses. The nurse-end program includes three functional modules: “Consultation”, “Management”, and “My”. The “Consultation” module is mainly used for online communication between case management nurses and patients. Nurses can enter the patient’s name in the search box to open a dialog box, and communicate with each other by sending text, voice and pictures. In the “Management” module, nurses can effortlessly search for patients by entering their name, WeChat nickname, or mobile phone number in the search box. This initiates a seamless dialogue, and with a simple click of the “+” button, patients can be promptly added to the “My Concerns” list. They can view the medical record information on its homepage, and add the postoperative treatment plan for the patient. The “self-management report” feature empowers nurses to stay up-to-date with patients’ recent well-being. By monitoring vital indicators like temperature, weight, and incidents of nausea or vomiting following chemotherapy, nurses can proactively ensure patients’ safety. The “clock in record” feature meticulously logs various patient activities including weight variations, exercise regimens, and medication adherence, providing a holistic view of their health journey. “Treatment monitoring Schedule” enables nurses to create customized chemotherapy schedules. With the first postoperative chemotherapy session scheduled in the calendar, the system seamlessly computes subsequent chemotherapy sessions and associated assessments. This transition to an online system marks a significant advancement from the traditional paper-based chemotherapy planning. Its automated scheduling and data tracking functions serve to alleviate the clinical nursing workload, enhancing efficiency and freeing up valuable time for focused patient care. The “My” module offers nurses the convenience of adding patients of interest or relevant content to their “My Favorites” section, enabling streamlined one-click access for viewing and management. The core structure and informational components of this module are outlined in Fig.  5 .

The main menu of nurse-end program

Driving the development process via the multi-stage optimization strategy

We adopted a multi-phase optimization strategy to drive the software development process. This strategy was proposed by Collins in 2005 and has become an important guiding theory for the development and evaluation of M-health interventions in recent years [ 40 ]. The strategy consists of three phases: Screening Phase, Refining Phase, and Confirming Phase. The Screening Phase need theories to identify and incorporate intervention elements. In this study, the initial version (1.0) development was based on self-management theory. Focusing on self-management, the results of contextual analysis, literature review and expert consultation were combined to design the mini-program version (1.0). The Refining Phase involves iterative adjustments to the previously version. In this study, the development team iteratively adjusted the mini-program version (1.0) according to users’ suggestions and test results. The Confirming Phase includes planning for clinical trials to test effect of the mini-program version (2.0) on self-management and recovery outcomes in out-of-hospital breast cancer patients.

Results of system test

Eight out-of-hospital breast cancer patients were recruited for system tests. The patient’s general information is listed in Table  2 .

The 10-item System Availability Scale (SUS)developed by Brooke was used [ 41 ]. The scale is a widely used method for quantitatively assessing user satisfaction with software systems. SUS is a Likert-5 and 10-item questionnaire (4 = strongly agree, 0 = strongly disagree), with Cronbach Alpha of 0.91. Generally, a system score above 60 on the SUS scale could be considered to be easy and simple to use, and the average score of SUS in our research is 78.75. The SUS scores of the mini-program system are presented in Fig.  6 .

System availability scale (SUS) score of patients

The research team designed the core task tests based on the typical and necessary self-management tasks of out-of-hospital patients. The core task of the “Information” module was listed as an example (Table  3 ). Functional tests include the passing rate for each task, and performance tests include the completion time of each task. More details can be found in Table  4 .

In this article, we demonstrated how to create a customized software solution for breast cancer case management practices based on a multi-stage optimization strategy, applied the contextual analysis method, and followed the user-centered principle. Preliminary test results showed satisfaction and acceptance of the WeChat mini-program among both out-of-hospital breast cancer patients and oncology nursing specialists.

Team effort

There were two typical patterns for developing M-health applications in the past. One was led by software developers, while the other was led by medical professionals. Each of these patterns has its own advantages and disadvantages. To overcome these shortcomings, some projects [ 42 ] developing M-health applications are now utilizing interdisciplinary team collaborations. This approach not only ensures the quality of the software but also makes sure that applications meet the actual needs.

In order to develop a customized software solution, our research team consisted of researchers, oncology nursing specialists, and software developers. The interdisciplinary team work dedicated to customizing software solutions together. Our team members each played to their strengths and held regular meetings to discuss and enhance our understanding and resolution of issues encountered during the software development process. Our team also included informal members: breast cancer patients, whose suggestions contributed to the practicality of the program.

Contextual analysis and user-centered design

Contextual analysis is a valuable tool that enables developers to design systems that are more relevant and user-friendly. And it allows us to understand any context-specific characteristics, practice patterns, and the openness of the target setting’s nurses and patients towards technology [ 42 ]. User-centered design can significantly reduce the cost of program iteration. More importantly, it has a profound influence on various aspects of a program including its design, functionality, information architecture, and interactive elements [ 43 ]. By analyzing different contexts, not only did we design features that better meet user needs, but we also predicted and addressed potential issues that users may encounter when using the mini-program in advance, thereby enhancing the user experience. In the iterative development stage, we discovered and improved some deficiencies in the design through core task testing and usability testing. Notably, the completion rate of the core task test reached 100%, indicating that our application is user-friendly and easy to operate.

• Multi-stage optimization strategy

In several priority areas of public health, researchers have successfully applied multi-stage optimization strategies to enhance their work, including software development and intervention programs [ 44 , 45 , 46 ]. In this study, we also apply this strategy to software development. While the multi-stage optimization strategy provides an optimization framework, it is important to note that our optimization objectives (such as software functionality and content requirements) are determined by key users involved in the research (out-of-hospital breast cancer patients and oncology nurse spescialists). This project adopts a multi-stage optimization strategy, iteratively improving the development of the mini-program through screening, refinement, and confirmation stages. Each stage aims to optimize our program.

The research team plans to explore the feasibility of mini program development program through preliminary experiment, and verify the intervention effect of mini program on self-management behavior, self-efficacy and quality of life and other indicators through formal experiment. A randomized controlled trial (IRB-2020-408) was initiated in August 2022 at a Class III hospital in Zhejiang, China, and is currently in the data collection phase.

There is no doubt that M-health will play a core role in the future of health care. However, to successfully implement and promote M-health applications in clinical setting, it is essential to analyze the needs of the target population. Additionally, it is crucial to determine who will be the driving force behind the implementation of the entire M-health project. This study demonstrates how to integrate M-health components into existing breast cancer case management care practices. In addition to providing a reference for other teams interested in developing and integrating M-health components into case management care models, this study also provides a reference for building M-health-featured care work models in practices.

In this study, the collaborative work of an interdisciplinary team with backgrounds in nursing and computer science, along with the active involvement of patients, not only facilitated the planning, developing, updating, and testing of M-health components based on the actual needs of the target population, but also increased the chances of acceptance and long-term implementation of the M-health program in practice.

This study demonstrates how to integrate M-health components into existing breast cancer case management practices. It provides insights for other reserch teams interested in developing and integrating M-health components into daily nursingt practice.

In the context of the digital age, M-health applications are rapidly becoming information sources and decision support tools for healthcare professionals and patients. However, it is crucial not to overlook the issues of information security and digital barriers for older adults.

Through interviews with outpatients with breast cancer and oncology nurses, we have gained insights into their concerns regarding information security. Some interviewees expressed concerns about information security and were worried about the risk of their personal information being leaked during app usage. Such concerns, to some extent, hinder the widespread adoption of M-health applications. Additionally, some interviewees mentioned that older patients, in general, find it challenging to learn and use the various functions of WeChat mini-programs, making it difficult to promote and apply M-health applications among the elderly population.

Solving these issues effectively is not only vital for the patients’ rights and interests but also crucial for the comprehensive implementation of M-health in practice. It is a matter that requires careful consideration in future development of M-health applications.

Data availability

The datasets generated and/or analysed during the current study are not publicly available but are available from the corresponding author on reasonable request.

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Acknowledgements

The authors would like to express our sincere gratitude to all the breast cancer patients who participated in this research.

This study was supported by the Zhejiang Provincial Natural Science Foundation of China (LY18H160061) and Funding for innovation and entrepreneurship of high-level overseas students in Hangzhou.

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Hong Chengang, Wang Liping, Wang Shujin, Chen Chen, Yang Jiayue, Lu Jingjing, Hua Shujie, Wu Jieming, Yao Liyan, Zeng Ni, Chu Jinhui & Sun Jiaqi

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HCG conceived the entire paper framework and was responsible for writing the paper. WSJ and CC conducted all interviews and managed the mini-programs. YJY, LJJ and HSJ were responsible for the collection of clinical nurse data. CJH and SJQ were responsible for patient data collection. Data analysis was conducted by WJM, YLY and ZN. WLP was responsible for the revision, editing and approval of manuscripts. All authors have rigorously revised and edited successive drafts of the manuscript. All authors read and approved the final version of the manuscript.

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Chengang, H., Liping, W., Shujin, W. et al. Multi-stage optimization strategy based on contextual analysis to create M-health components for case management model in breast cancer transitional care: the CMBM study as an example. BMC Nurs 23 , 385 (2024). https://doi.org/10.1186/s12912-024-02049-x

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Experiences of medical students and faculty regarding the use of long case as a formative assessment method at a tertiary care teaching hospital in a low resource setting: a qualitative study

• Jacob Kumakech 1 ,
• Ian Guyton Munabi 2 ,
• Aloysius Gonzaga Mubuuke 3 &
• Sarah Kiguli 4  

BMC Medical Education volume  24 , Article number:  621 ( 2024 ) Cite this article

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Introduction

The long case is used to assess medical students’ proficiency in performing clinical tasks. As a formative assessment, the purpose is to offer feedback on performance, aiming to enhance and expedite clinical learning. The long case stands out as one of the primary formative assessment methods for clinical clerkship in low-resource settings but has received little attention in the literature.

To explore the experiences of medical students and faculty regarding the use of the Long Case Study as a formative assessment method at a tertiary care teaching hospital in a low-resource setting.

Methodology

A qualitative study design was used. The study was conducted at Makerere University, a low-resource setting. The study participants were third- and fifth-year medical students as well as lecturers. Purposive sampling was utilized to recruit participants. Data collection comprised six Focus Group Discussions with students and five Key Informant Interviews with lecturers. The qualitative data were analyzed by inductive thematic analysis.

Three themes emerged from the study: ward placement, case presentation, and case assessment and feedback. The findings revealed that students conduct their long cases at patients’ bedside within specific wards/units assigned for the entire clerkship. Effective supervision, feedback, and marks were highlighted as crucial practices that positively impact the learning process. However, challenges such as insufficient orientation to the long case, the super-specialization of the hospital wards, pressure to hunt for marks, and inadequate feedback practices were identified.

The long case offers students exposure to real patients in a clinical setting. However, in tertiary care teaching hospitals, it’s crucial to ensure proper design and implementation of this practice to enable students’ exposure to a variety of cases. Adequate and effective supervision and feedback create valuable opportunities for each learner to present cases and receive corrections.

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The long case serves as an authentic assessment method for evaluating medical students’ competence in clinical tasks [ 1 ]. This form of assessment requires students to independently spend time with patients taking their medical history, conducting physical examinations, and formulating diagnosis and management plans. Subsequently, students present their findings to senior clinicians for discussion and questioning [ 2 , 3 ]. While developed countries increasingly adopt simulation-based assessments for formative evaluation, logistical challenges hinder the widespread use of such methods in developing countries [ 4 ]. Consequently, the low-resource countries heavily rely on real patient encounters for formative assessment. The long case is one such method predominantly used as a primary formative assessment method during clinical clerkship and offers a great opportunity for feedback [ 5 ]. The assessment grounds students’ learning into practice by providing them with rich opportunities to interact with patients and have the feel of medical practice. The long case thus bridges the gap between theory and practice, immersing students in the real tasks of a physician [ 1 ]. The complexity of clinical scenarios and the anxiety associated with patient encounters may not be well replicated in simulation-based assessments because diseases often have atypical presentations not found in textbooks. Assessment methods should thus utilize authentic learning experiences to provide learners with applications of learning that they would expect to encounter in real life [ 6 ]. This requires medical education and the curriculum to focus attention on assessment because it plays a significant role in driving learning [ 7 ]. The long case thus remains crucial in medical education as one of the best ways of preparing for practice. It exposes the student repeatedly to taking medical history, examining patients, making clinical judgments, deciding treatment plans, and collaborating with senior clinicians.

The long case, however, has faced significant criticism in the medical education literature due to perceived psychometric deficiencies [ 8 , 9 , 10 ]. Consequently, many universities have begun to adopt assessment methods that yield more reliable and easily defensible results [ 2 ] due to concerns over the low reliability, generalizability, and validity of the long case, coupled with rising litigations and student appeals [ 11 , 12 ]. Despite these shortcomings, the long case remains an educationally valuable assessment tool that provides diagnostic feedback essential for the learning process during clinical clerkship [ 13 ]. Teachers can utilize long-case results to pinpoint neglected areas or teaching deficiencies and align with course outcomes.

However, there is a paucity of research into the long case as a formative assessment tool. A few studies conducted in developed countries highlighted its role in promoting a holistic approach to patient care, fostering students’ clinical skills, and a driving force for students to spend time with patients [ 2 , 13 ], . There is a notable absence of literature on the use of long case as a formative assessment method in low-resource countries, and no published work is available at Makerere University where it has been used for decades. This underscores the importance of conducting research in this area to provide insight into the effectiveness, challenges, and potentials for improvement. Therefore, this study aimed to investigate the experiences of medical students and faculty regarding the utilization of the long case as a formative assessment method within the context of a tertiary care teaching hospital in a low-resource setting.

Study design

This was an exploratory qualitative study.

Study setting

The research was conducted at Makerere University within the Department of Internal Medicine. The Bachelor of Medicine and Bachelor of Surgery (MBChB) degree at Makerere University is a five-year program with the first two years for pre-clinical (biomedical Sciences) course and the last three years dedicated to clinical clerkship. Medical students do Internal Medicine clerkships in third- and fifth-year at the two tertiary teaching hospitals namely; Mulago and Kiruddu National Referral Hospitals. The students are introduced to the long case in third-year as Junior Clerks and later in the fifth-year as Senior Clerks. During clerkship, students are assigned to various medical wards, where they interact with patients, take medical history from them, perform physical examinations, and develop diagnosis and management plans. Subsequently, students present their long cases to lecturers or postgraduate students, often in the presence of their peers, followed by feedback and comprehensive case discussions. Students are afforded ample time to prepare and present their cases during ward rounds, at their discretion. The students are formatively assessed and a mark is awarded on a scale of one to ten in the student’s logbook. Each student is required to make a minimum of ten long cases over the seven weeks of clerkship.

Study participants

The study participants were third- and fifth-year medical students who had completed junior and senior clerkship respectively, as well as lecturers who possessed at least five years of experience with the long case. The participants were selected through purposive sampling. The sample size for the study was determined by data saturation.

Data collection

Data were collected through Focus Group Discussions (FGDs) and Key Informant Interviews (KIIs). A total of 36 medical students participated in FGDs, reflecting on their experiences with the long case. Five faculty members participated in individual KIIs. The students were mobilized by their class representative and a brief recruitment presentation was made at the study site while the lecturers were approached via email and telephone invitation.

Six FGDs were conducted, three for junior clerks and three for senior clerks. Each FGD comprised of 5–7 participants with balanced male and female gender representation. Data saturation was achieved by the fifth FGD, at which point no additional new information emerged. A research assistant proficient in qualitative research methods moderated the FGDs. The discussions lasted between 55 min and 1 h 10 min and were audio recorded. The Principal Investigator attended all the FGDs to document interactions and record his perspectives and non-verbal cues of participants.

Semi-structured KIIs were used to collect data from Internal Medicine faculty. Five KIIs were conducted, and data saturation was achieved by the fourth interview, at which point no new theme emerged. The Principal Investigator conducted the KIIs via Zoom. Each interview lasted between 25 and 50 min and all were audio recorded. A research assistant proficient in qualitative methods attended all the Zoom meetings. The data collected were securely stored on a hard drive and Google Drive with password protection to prevent unauthorized access.

Data analysis

Data analysis was done through inductive thematic analysis method. Following each FGD or KII session, the data collection team listened to the recordings to familiarize themselves with the data and develop general ideas regarding the participants’ perspectives. The data were transcribed verbatim by the researchers to generate text data. Two separate transcripts were generated by the Principal Investigator and a research assistant. The transcripts were then compared and manually reviewed by the research team to compare the accuracy with the audio recordings. After transcript harmonization, data cleaning was done for both FGDs and KIIs transcripts.

The transcribed data from both FGDs and KIIs underwent inductive thematic analysis as aggregated data. This involved initial line-by-line coding, followed by focused coding where the relationships between initial codes were explored and similar codes were grouped. Throughout the analysis, the principle of constant comparison was applied, where emerging codes were compared for similarities and differences.

Study results

Socio-demographics.

A total of 36 medical students participated in the FGDs, comprising 18 junior clerks and 19 senior clerks. The participants were aged between 21 and 25 years except two participants who were aged above 25 (30 and 36 years old). Among the third-year students, there were 10 male and 9 female participants while the fifth-year student comprised of 8 male and 10 female participants.

Five lecturers participated in the Key Informant Interviews, three of whom were females and two male participants. They were aged between 40 and 50 years, and all had over 10 years of experience with the long case. The faculty members included one consultant physician, one associate professor, two senior lecturers, and one lecturer.

Themes that emerged

Three themes emerged from the study: ward placement, case presentations, and case assessment and feedback.

Theme 1: Ward placement

The study findings disclosed that medical students are assigned to specific wards for the duration of their clerkship. The specialization of medical wards was found to significantly restrict students’ exposure to limited disease conditions found only in their allocated ward.

With the super-specialization of the units, there is some bias on what they do learn; if a particular group is rotating on the cardiology unit, they will obviously have a bias to learn the history and physical exam related to cardiovascular disease (KII 1).

The students, particularly junior clerks, expressed dissatisfaction with the lack of proper and standardized orientation to the long case on the wards. This deficiency led to wastage of time and a feeling of being unwelcome in the clerkship.

Some orient you when you reach the ward but others you reach and you are supposed to pick up on your own. I expect orientation, then taking data from us, what they expect us to do, and what we expect from them, taking us through the clerkship sessions (FGD 4 Participant 1).

Students’ exposure to cases in other wards poses significant challenges; the study found that as some lecturers facilitate visits to different wards for scheduled teaching sessions, others don’t, resulting in missed learning opportunities. Additionally, some lecturers leave the burden on students’ personal initiative to explore cases in other wards.

We actually encourage them to go through the different specialties because when you are faced with a patient, you will not have to choose which one to see and not to see (KII 4).

Imagine landing on a stroke patient when you have been in the infectious disease ward or getting a patient with renal condition when you have been in the endocrinology ward can create problems (FGD 6 Participant 3).

Theme 2 Case presentation

Medical students present their long case to lecturers and postgraduate students. However, participants revealed variations among lecturers regarding their preferences on how they want students to present their cases. While some prefer to listen to the entire history and examination, others prefer only a summary, and some prefer starting from the diagnosis.

The practice varies depending on the lecturer, as everyone does it their own way. There are some, who listen to your history, examination, and diagnosis, and then they go into basic discussion of the case; others want only a summary. Some lecturers come and tell you to start straight away from your diagnosis, and then they start treating you backward (FGD 6 Participant 3).

The students reported limited observation of their skills due a little emphasis placed by examiners on physical examination techniques, as well as not providing the students with the opportunity to propose treatment plans.

When we are doing these physical examinations on the ward no one is seeing you. You present your physical examination findings, but no one saw how you did it. You may think you are doing the right thing during the ward rotations, but actually your skills are bad (FGD 4 Participant 6).

They don’t give us time to propose management plans. The only time they ask for how you manage a patient is during the summative long case, yet during the ward rotation, they were not giving us the freedom to give our opinion on how we would manage the patient.(FGD 2Participant 6).

Supervision was reportedly dependent on the ward to which the student was allocated. Additionally, the participants believe that the large student-to-lecturer ratio negatively affects the opportunity to present.

My experience was different in years three and five. In year three, we had a specialist every day on the ward, but in year five, we would have a specialist every other day, sometimes even once a week. When I compare year five with year three, I think I was even a better doctor in year three than right now (FGD 1 Participant 1).

Clinical training is like nurturing somebody to behave or conduct themselves in a certain way. Therefore, if the numbers are large, the impacts per person decrease, and the quality decreases (KII 5).

Theme C: Case assessment and feedback

The study found that a student’s long case is assessed both during the case presentation on the ward and through the case write-up, with marks awarded accordingly.

They present to the supervisor and then also write it up, so at a later time you also mark the sheet where they have written up the cases; so they are assessed at presentation and write up (KII 2).

The mark awarded was reportedly a significant motivator for students to visit wards and clerk patients, but students also believe that the pressure to hunt for marks tends to override the goal of the formative assessment.

Your goal there is to learn, but most of us go with the goal of getting signatures; signature-based learning. The learning, you realize probably comes on later if you have the individual morale to go and learn (FGD 1 participant 1).

Feedback is an integral part of any formative assessment. While students receive feedback from lecturers, the participants were concerned about the absence of a formal channel for soliciting feedback from students.

Of course, teachers provide feedback to students because it is a normal part of teaching. However, it is not a common routine to solicit feedback about how teaching has gone. So maybe that is something that needs to be improved so that we know if we have been effective teachers (KII 3).

Whereas the feedback intrigues students to read more to compensate for their knowledge gap, they decried several encounters with demeaning, intimidating, insulting, demotivating, and embarrassing feedback from assessors.

Since we are given a specific target of case presentation we are supposed to make in my training , if I make the ten, I wouldn’t want to present again. Why would I receive other negative comments for nothing? They truly have a personality effect on the student, and students feel low self-esteem (FGD 1, Participant 4).

This study aimed to investigate the experiences of medical students and faculty regarding the use of the long case as a formative assessment method at a tertiary care teaching hospital in a low-resource setting. This qualitative research provides valuable insights into the current practices surrounding the long case as a formative assessment method in such a setting.

The study highlighted the patient bedside as the primary learning environment for medical students. Bedside teaching plays a crucial role in fostering the development of skills such as history-taking and physical examination, as well as modeling professional behaviors and directly observing learners [ 14 , 15 ]. However, the specialization of wards in tertiary hospitals means that students may not be exposed to certain conditions found in other wards. This lack of exposure can lead to issues of case specificity, which has been reported in various literature as a cause of low reliability and generalizability of the long case [ 16 , 17 ]. Participants in the study expressed feeling like pseudo-specialists based on their ward allocations. This is partly attributed to missing scheduled teachings and poor management of opportunities to clerk and present patients on other wards. Addressing these challenges is essential for enhancing the effectiveness of the long case as a formative assessment method in medical education.

Proper orientation at the beginning of a clerkship is crucial for clarifying the structure and organization, defining students’ roles, and providing insights into clinical supervisors’ perspectives [ 18 ]. However, the study revealed that orientation into the long case was unsatisfactory, resulting in time wastage and potentially hindering learning. Effective orientation requires dedicated time and should involve defining expectations and goals, as well as guiding students through the steps of history-taking and physical examination during the initial weeks of the rotation. Contrary to this ideal approach, the medical students reported being taken through systemic examinations when the clerkship was nearing its end, highlighting a significant gap in the orientation process. Proper orientation is very important since previous studies have also documented the positive impact of orientation on student performance [ 19 ]. Therefore, addressing the shortcomings in orientation practices identified in this study is essential for optimizing learning outcomes and ensuring that students are adequately prepared to engage in the long case.

There was reportedly a significant variation in the way students present their long cases, with some lecturers preferring only a case summary, while others expect a complete presentation or begin with a diagnosis. While this diversity in learning styles may expose students to both familiar and unfamiliar approaches, providing a balance of comfort and tension [ 20 ], it’s essential for students to first be exposed to familiar methods before transitioning to less familiar ones to expand their ability to use diverse learning styles. The variation observed in this context may be attributed to time constraints, as lecturers may aim to accommodate the large number of students within the available time. Additionally, a lack of standardized practices could also contribute to this variation. Therefore, there is a pressing need for standardized long-case practices to ensure a consistent experience for students and to meet the desired goals of the assessment. Standardizing the long case practice would not only provide a uniform experience for students but also enhance the reliability, validity, and perception of fairness of the assessment [ 9 , 21 ]. It would ensure that all students are evaluated using the same criteria, reducing potential biases and disparities in grading. Additionally, standardized practices facilitate better alignment with learning objectives and promote more effective feedback mechanisms [ 22 ].

Related to the above, students reported limited observation of skills and little emphasis placed on them to learn physical examination techniques. This finding resonates with the research conducted by Abdalla and Shorbagi in 2018, where many students reported a lack of observation during history-taking and physical examination [ 23 ]. The importance of observation is underscored by the fact that students often avoid conducting physical examinations, as highlighted in Pavlakis & Laurent’s study among postgraduate trainees in 2001 [ 24 ]. This study sheds more light on the critical role of observation in forcing medical students to master clinical assessment and practical skills. The study also uncovered that students are rarely given the opportunity to propose management plans during case presentations, which hampers their confidence and learning of clinical decision-making. These findings likely stem from the large student-to-lecturer ratio and little attention given to these aspects of the long case during the planning of the assessment method. The result is students not receiving the necessary guidance and support to develop their clinical and decision-making skills. Therefore, addressing these issues by putting more emphasis on observation of student-patient interaction, management plan, and having a smaller student group is vital to ensure that medical students receive comprehensive training and are adequately prepared for their future roles as physicians.

The study found that the marks awarded for the long case serve as the primary motivator for students. This finding aligns with previous research indicating that the knowledge that each long case is part of assessment drives students to perform their duties diligently [ 2 , 25 ]. It underscores the crucial role that assessment plays in driving learning processes. However, the pressures to obtain marks and signatures reportedly hinder students’ engagement in learning. This could be attributed to instances where some lecturers relax on supervision or are absent, leaving students to struggle to find someone to assess them. Inadequate supervision by attending physicians has been identified in prior studies as one of the causes of insufficient clinical experience [ 26 ], something that need to be dealt with diligently. While the marks awarded are a motivating factor, it is essential to understand other underlying motivations of medical students to engage in the long case and their impact on the learning process.

Feedback is crucial for the long case to fulfill its role as an assessment for learning. The study participants reported that feedback is provided promptly as students present their cases. This immediate feedback is essential for identifying errors and learning appropriate skills to enhance subsequent performance. However, the feedback process appears to be unilateral, with students receiving feedback from lecturers but lacking a structured mechanism for providing feedback themselves. One reason for the lack of student feedback may be a perceived intimidating approach from lecturers which discourages students from offering their input. It is thus important to establish a conducive environment where students feel comfortable providing feedback without fear of negative repercussions. The study underscores the significance of feedback from students in improving the learning process. This aligns with the findings of Hattie and Timperley (2007), who emphasized that feedback received from learners contributes significantly to improvements in student learning [ 27 ]. Therefore, it is essential to implement strategies to encourage and facilitate bidirectional feedback between students and lecturers in the context of the long case assessment. This could involve creating formal channels for students to provide feedback anonymously or in a structured format, fostering open communication, and addressing any perceived barriers to feedback exchange [ 28 ]. By promoting a culture of feedback reciprocity, educators can enhance the effectiveness of the long case as an assessment tool.

Conclusions

In conclusion, the long case remains a cornerstone of formative assessment during clerkship in many medical schools, particularly in low-resource countries. However, its effectiveness is challenged by limitations such as case specificity in tertiary care hospitals, which can affect the assessment’s reliability and generalizability. The practice of awarding marks in formative assessment serves as a strong motivator for students but also creates tension, especially when there is inadequate contact with lecturers. This can lead to a focus on hunting for marks at the expense of genuine learning. Thus adequate supervision and feedback practices are vital for ensuring the success of the long case as an assessment for learning.

Furthermore, there is a need to foster standardized long case practice to ensure that scheduled learning activities are completed and that all students clerk and present patients with different conditions from various wards. This will promote accountability among both lecturers and students and ensure a consistent and uniform experience with the long case as an assessment for learning, regardless of the ward a student is assigned.

Data availability

The data supporting the study results of this article can be accessed from the Makerere University repository, titled “Perceptions of Medical Students and Lecturers of the Long Case Practices as Formative Assessment in Internal Medicine Clerkship at Makerere University,” available on DSpace. The identifier is http://hdl.handle.net/10570/13032 . Additionally, the raw data are securely stored with the researchers in Google Drive.

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This research was supported by the Fogarty International Centre of the National Institute of Health under award number 1R25TW011213. The content is solely the responsibility of the author and does not necessarily represent the official views of the National Institute of Health.

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Jacob Kumakech

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School of Medicine, Department of Radiology, Makerere University, Kampala, Uganda

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JK contributed to the conception and design of the study, as well as the acquisition, analysis, and interpretation of the data. He also drafted the initial version of the work and approved the submitted version. He agrees to be personally accountable for his contribution and to ensure that any questions related to the accuracy or integrity of any part of the work, even those in which he was not personally involved, are appropriately investigated and resolved, with the resolution documented in the literature.IMG contributed to the analysis and interpretation of the data. He also made major corrections to the first draft of the manuscript and approved the submitted version. He agrees to be personally accountable for his contribution and to ensure that any questions related to the accuracy or integrity of any part of the work, even those in which he was not personally involved, are appropriately investigated and resolved, with the resolution documented in the literature.MA contributed to the analysis and interpretation of the data. He made major corrections to the first draft of the manuscript and approved the submitted version. He agrees to be personally accountable for his contribution and to ensure that any questions related to the accuracy or integrity of any part of the work, even those in which he was not personally involved, are appropriately investigated and resolved, with the resolution documented in the literature.SK made major corrections to the first draft and the final corrections for the submitted version of the work. She agrees to be personally accountable for her contribution and to ensure that any questions related to the accuracy or integrity of any part of the work, even those in which she was not personally involved, are appropriately investigated and resolved, with the resolution documented in the literature.

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Correspondence to Jacob Kumakech .

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Ethical approval to conduct the study was obtained from the Makerere University School of Medicine Research and Ethics Committee, with ethics ID Mak-SOMREC-2022-524. Informed consent was obtained from all participants using the Mak-SOMREC informed consent form.

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Kumakech, J., Munabi, I.G., Mubuuke, A.G. et al. Experiences of medical students and faculty regarding the use of long case as a formative assessment method at a tertiary care teaching hospital in a low resource setting: a qualitative study. BMC Med Educ 24 , 621 (2024). https://doi.org/10.1186/s12909-024-05589-7

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Received : 04 April 2024

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Published : 05 June 2024

DOI : https://doi.org/10.1186/s12909-024-05589-7

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Long COVID or Post-COVID Conditions

Some people who have been infected with the virus that causes COVID-19 can experience long-term effects from their infection, known as Long COVID or Post-COVID Conditions (PCC). Long COVID is broadly defined as signs, symptoms, and conditions that continue or develop after acute COVID-19 infection. This definition  of Long COVID was developed by the Department of Health and Human Services (HHS) in collaboration with CDC and other partners.

People call Long COVID by many names, including Post-COVID Conditions, long-haul COVID, post-acute COVID-19, long-term effects of COVID, and chronic COVID. The term post-acute sequelae of SARS CoV-2 infection (PASC) is also used to refer to a subset of Long COVID.

What You Need to Know

• Long COVID is a real illness and can result in chronic conditions that require comprehensive care. There are resources available .
• Long COVID can include a wide range of ongoing health problems; these conditions can last weeks, months, or years.
• Long COVID occurs more often in people who had severe COVID-19 illness, but anyone who has been infected with the virus that causes COVID-19 can experience it.
• People who are not vaccinated against COVID-19 and become infected may have a higher risk of developing Long COVID compared to people who have been vaccinated.
• People can be reinfected with SARS-CoV-2, the virus that causes COVID-19, multiple times. Each time a person is infected or reinfected with SARS-CoV-2, they have a risk of developing Long COVID.
• While most people with Long COVID have evidence of infection or COVID-19 illness, in some cases, a person with Long COVID may not have tested positive for the virus or known they were infected.
• CDC and partners are working to understand more about who experiences Long COVID and why, including whether groups disproportionately impacted by COVID-19 are at higher risk.

In July 2021, Long COVID was added as a recognized condition that could result in a disability under the Americans with Disabilities Act (ADA). Learn more: Guidance on “Long COVID” as a Disability Under the ADA .

About Long COVID

Long COVID is a wide range of new, returning, or ongoing health problems that people experience after being infected with the virus that causes COVID-19. Most people with COVID-19 get better within a few days to a few weeks after infection, so at least 4 weeks after infection is the start of when Long COVID could first be identified. Anyone who was infected can experience Long COVID. Most people with Long COVID experienced symptoms days after first learning they had COVID-19, but some people who later experienced Long COVID did not know when they got infected.

There is no test that determines if your symptoms or condition is due to COVID-19. Long COVID is not one illness. Your healthcare provider considers a diagnosis of Long COVID based on your health history, including if you had a diagnosis of COVID-19 either by a positive test or by symptoms or exposure, as well as based on a health examination.

Science behind Long COVID

RECOVER: Researching COVID to Enhance Recovery

People with Long COVID may experience many symptoms.

People with Long COVID can have a wide range of symptoms that can last weeks, months, or even years after infection. Sometimes the symptoms can even go away and come back again. For some people, Long COVID can last weeks, months, or years after COVID-19 illness and can sometimes result in disability.

Long COVID may not affect everyone the same way. People with Long COVID may experience health problems from different types and combinations of symptoms that may emerge, persist, resolve, and reemerge over different lengths of time. Though most patients’ symptoms slowly improve with time, speaking with your healthcare provider about the symptoms you are experiencing after having COVID-19 could help determine if you might have Long COVID.

People who experience Long COVID most commonly report:

General symptoms ( Not a Comprehensive List)

• Tiredness or fatigue that interferes with daily life
• Symptoms that get worse after physical or mental effort (also known as “ post-exertional malaise ”)

Respiratory and heart symptoms

• Difficulty breathing or shortness of breath
• Fast-beating or pounding heart (also known as heart palpitations)

Neurological symptoms

• Difficulty thinking or concentrating (sometimes referred to as “brain fog”)
• Sleep problems
• Dizziness when you stand up (lightheadedness)
• Pins-and-needles feelings
• Change in smell or taste
• Depression or anxiety

Digestive symptoms

• Stomach pain

Other symptoms

• Joint or muscle pain
• Changes in menstrual cycles

Symptoms that are hard to explain and manage

Some people with Long COVID have symptoms that are not explained by tests or easy to manage.

People with Long COVID may develop or continue to have symptoms that are hard to explain and manage. Clinical evaluations and results of routine blood tests, chest X-rays, and electrocardiograms may be normal. The symptoms are similar to those reported by people with myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) and other poorly understood chronic illnesses that may occur after other infections. People with these unexplained symptoms may be misunderstood by their healthcare providers, which can result in a delay in diagnosis and receiving the appropriate care or treatment.

Review these tips to help prepare for a healthcare provider appointment for Long COVID.

Health conditions

Some people experience new health conditions after COVID-19 illness.

Some people, especially those who had severe COVID-19, experience multiorgan effects or autoimmune conditions with symptoms lasting weeks, months, or even years after COVID-19 illness. Multi-organ effects can involve many body systems, including the heart, lung, kidney, skin, and brain. As a result of these effects, people who have had COVID-19 may be more likely to develop new health conditions such as diabetes, heart conditions, blood clots, or neurological conditions compared with people who have not had COVID-19.

People experiencing any severe illness may develop health problems

People experiencing any severe illness, hospitalization, or treatment may develop problems such as post-intensive care syndrome (PICS).

PICS refers to the health effects that may begin when a person is in an intensive care unit (ICU), and which may persist after a person returns home. These effects can include muscle weakness, problems with thinking and judgment, and symptoms of post-traumatic stress disorder  (PTSD), a long-term reaction to a very stressful event. While PICS is not specific to infection with SARS-CoV-2, it may occur and contribute to the person’s experience of Long COVID. For people who experience PICS following a COVID-19 diagnosis, it is difficult to determine whether these health problems are caused by a severe illness, the virus itself, or a combination of both.

People More Likely to Develop Long COVID

Some people may be more at risk for developing Long COVID.

Researchers are working to understand which people or groups of people are more likely to have Long COVID, and why. Studies have shown that some groups of people may be affected more by Long COVID. These are examples and not a comprehensive list of people or groups who might be more at risk than other groups for developing Long COVID:

• People who have experienced more severe COVID-19 illness, especially those who were hospitalized or needed intensive care.
• People who had underlying health conditions prior to COVID-19.
• People who did not get a COVID-19 vaccine.

Health Inequities May Affect Populations at Risk for Long COVID

Some people are at increased risk of getting sick from COVID-19 because of where they live or work, or because they can’t get health care. Health inequities may put some people from racial or ethnic minority groups and some people with disabilities at greater risk for developing Long COVID. Scientists are researching some of those factors that may place these communities at higher risk of getting infected or developing Long COVID.

Preventing Long COVID

The best way to prevent Long COVID is to protect yourself and others from becoming infected. For people who are eligible, CDC recommends staying up to date on COVID-19 vaccination , along with improving ventilation, getting tested for COVID-19 if needed, and seeking treatment for COVID-19 if eligible. Additional preventative measures include avoiding close contact with people who have a confirmed or suspected COVID-19 illness and washing hands  or using alcohol-based hand sanitizer.

Research suggests that people who get a COVID-19 infection after vaccination are less likely to report Long COVID, compared to people who are unvaccinated.

CDC, other federal agencies, and non-federal partners are working to identify further measures to lessen a person’s risk of developing Long COVID. Learn more about protecting yourself and others from COVID-19 .

Living with Long COVID

Living with Long COVID can be hard, especially when there are no immediate answers or solutions.

People experiencing Long COVID can seek care from a healthcare provider to come up with a personal medical management plan that can help improve their symptoms and quality of life. Review these tips  to help prepare for a healthcare provider appointment for Long COVID. In addition, there are many support groups being organized that can help patients and their caregivers.

Although Long COVID appears to be less common in children and adolescents than in adults, long-term effects after COVID-19 do occur in children and adolescents .

Talk to your doctor if you think you or your child has Long COVID. Learn more: Tips for Talking to Your Healthcare Provider about Post-COVID Conditions

Data for Long COVID

Studies are in progress to better understand Long COVID and how many people experience them.

CDC is using multiple approaches to estimate how many people experience Long COVID. Each approach can provide a piece of the puzzle to give us a better picture of who is experiencing Long COVID. For example, some studies look for the presence of Long COVID based on self-reported symptoms, while others collect symptoms and conditions recorded in medical records. Some studies focus only on people who have been hospitalized, while others include people who were not hospitalized. The estimates for how many people experience Long COVID can be quite different depending on who was included in the study, as well as how and when the study collected information.  Estimates of the proportion of people who had COVID-19 that go on to experience Long COVID can vary.

CDC posts data on Long COVID and provides analyses, the most recent of which can be found on the U.S. Census Bureau’s Household Pulse Survey .

CDC and other federal agencies, as well as academic institutions and research organizations, are working to learn more about the short- and long-term health effects associated with COVID-19 , who gets them and why.

Scientists are also learning more about how new variants could potentially affect Long COVID. We are still learning to what extent certain groups are at higher risk, and if different groups of people tend to experience different types of Long COVID. CDC has several studies that will help us better understand Long COVID and how healthcare providers can treat or support patients with these long-term effects. CDC will continue to share information with healthcare providers to help them evaluate and manage these conditions.

CDC is working to:

• Better identify the most frequent symptoms and diagnoses experienced by patients with Long COVID.
• Better understand how many people are affected by Long COVID, and how often people who are infected with COVID-19 develop Long COVID
• Better understand risk factors and protective factors, including which groups might be more at risk, and if different groups experience different symptoms.
• Help understand how Long COVID limit or restrict people’s daily activity.
• Help identify groups that have been more affected by Long COVID, lack access to care and treatment for Long COVID, or experience stigma.
• Better understand the role vaccination plays in preventing Long COVID.
• Collaborate with professional medical groups to develop and offer clinical guidance and other educational materials for healthcare providers, patients, and the public.

Related Pages

• Caring for People with Post-COVID Conditions
• Preparing for Appointments for Post-COVID Conditions
• Researching COVID to Enhance Recovery
• Guidance on “Long COVID” as a Disability Under the ADA

For Healthcare Professionals

• Post-COVID Conditions: Healthcare Providers

Search for and find historical COVID-19 pages and files. Please note the content on these pages and files is no longer being updated and may be out of date.

• Visit archive.cdc.gov for a historical snapshot of the COVID-19 website, capturing the end of the Federal Public Health Emergency on June 28, 2023.
• Visit the dynamic COVID-19 collection  to search the COVID-19 website as far back as July 30, 2021.

To receive email updates about COVID-19, enter your email address:

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• The Centers for Disease Control and Prevention (CDC) cannot attest to the accuracy of a non-federal website.
• Linking to a non-federal website does not constitute an endorsement by CDC or any of its employees of the sponsors or the information and products presented on the website.
• You will be subject to the destination website's privacy policy when you follow the link.
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