literature review in case study

Have a language expert improve your writing

Run a free plagiarism check in 10 minutes, generate accurate citations for free.

• Knowledge Base

Methodology

• How to Write a Literature Review | Guide, Examples, & Templates

How to Write a Literature Review | Guide, Examples, & Templates

Published on January 2, 2023 by Shona McCombes . Revised on September 11, 2023.

What is a literature review? A literature review is a survey of scholarly sources on a specific topic. It provides an overview of current knowledge, allowing you to identify relevant theories, methods, and gaps in the existing research that you can later apply to your paper, thesis, or dissertation topic .

There are five key steps to writing a literature review:

• Search for relevant literature
• Evaluate sources
• Identify themes, debates, and gaps
• Outline the structure
• Write your literature review

A good literature review doesn’t just summarize sources—it analyzes, synthesizes , and critically evaluates to give a clear picture of the state of knowledge on the subject.

Instantly correct all language mistakes in your text

Upload your document to correct all your mistakes in minutes

Table of contents

What is the purpose of a literature review, examples of literature reviews, step 1 – search for relevant literature, step 2 – evaluate and select sources, step 3 – identify themes, debates, and gaps, step 4 – outline your literature review’s structure, step 5 – write your literature review, free lecture slides, other interesting articles, frequently asked questions, introduction.

• Quick Run-through
• Step 1 & 2

When you write a thesis , dissertation , or research paper , you will likely have to conduct a literature review to situate your research within existing knowledge. The literature review gives you a chance to:

• Demonstrate your familiarity with the topic and its scholarly context
• Develop a theoretical framework and methodology for your research
• Position your work in relation to other researchers and theorists
• Show how your research addresses a gap or contributes to a debate
• Evaluate the current state of research and demonstrate your knowledge of the scholarly debates around your topic.

Writing literature reviews is a particularly important skill if you want to apply for graduate school or pursue a career in research. We’ve written a step-by-step guide that you can follow below.

Receive feedback on language, structure, and formatting

Professional editors proofread and edit your paper by focusing on:

• Academic style
• Vague sentences
• Style consistency

See an example

Writing literature reviews can be quite challenging! A good starting point could be to look at some examples, depending on what kind of literature review you’d like to write.

• Example literature review #1: “Why Do People Migrate? A Review of the Theoretical Literature” ( Theoretical literature review about the development of economic migration theory from the 1950s to today.)
• Example literature review #2: “Literature review as a research methodology: An overview and guidelines” ( Methodological literature review about interdisciplinary knowledge acquisition and production.)
• Example literature review #3: “The Use of Technology in English Language Learning: A Literature Review” ( Thematic literature review about the effects of technology on language acquisition.)
• Example literature review #4: “Learners’ Listening Comprehension Difficulties in English Language Learning: A Literature Review” ( Chronological literature review about how the concept of listening skills has changed over time.)

You can also check out our templates with literature review examples and sample outlines at the links below.

Download Word doc Download Google doc

Before you begin searching for literature, you need a clearly defined topic .

If you are writing the literature review section of a dissertation or research paper, you will search for literature related to your research problem and questions .

Make a list of keywords

Start by creating a list of keywords related to your research question. Include each of the key concepts or variables you’re interested in, and list any synonyms and related terms. You can add to this list as you discover new keywords in the process of your literature search.

• Social media, Facebook, Instagram, Twitter, Snapchat, TikTok
• Body image, self-perception, self-esteem, mental health
• Generation Z, teenagers, adolescents, youth

Search for relevant sources

Use your keywords to begin searching for sources. Some useful databases to search for journals and articles include:

• Your university’s library catalogue
• Google Scholar
• Project Muse (humanities and social sciences)
• Medline (life sciences and biomedicine)
• EconLit (economics)
• Inspec (physics, engineering and computer science)

You can also use boolean operators to help narrow down your search.

Make sure to read the abstract to find out whether an article is relevant to your question. When you find a useful book or article, you can check the bibliography to find other relevant sources.

You likely won’t be able to read absolutely everything that has been written on your topic, so it will be necessary to evaluate which sources are most relevant to your research question.

For each publication, ask yourself:

• What question or problem is the author addressing?
• What are the key concepts and how are they defined?
• What are the key theories, models, and methods?
• Does the research use established frameworks or take an innovative approach?
• What are the results and conclusions of the study?
• How does the publication relate to other literature in the field? Does it confirm, add to, or challenge established knowledge?
• What are the strengths and weaknesses of the research?

Make sure the sources you use are credible , and make sure you read any landmark studies and major theories in your field of research.

You can use our template to summarize and evaluate sources you’re thinking about using. Click on either button below to download.

Take notes and cite your sources

As you read, you should also begin the writing process. Take notes that you can later incorporate into the text of your literature review.

It is important to keep track of your sources with citations to avoid plagiarism . It can be helpful to make an annotated bibliography , where you compile full citation information and write a paragraph of summary and analysis for each source. This helps you remember what you read and saves time later in the process.

The only proofreading tool specialized in correcting academic writing - try for free!

The academic proofreading tool has been trained on 1000s of academic texts and by native English editors. Making it the most accurate and reliable proofreading tool for students.

Try for free

To begin organizing your literature review’s argument and structure, be sure you understand the connections and relationships between the sources you’ve read. Based on your reading and notes, you can look for:

• Trends and patterns (in theory, method or results): do certain approaches become more or less popular over time?
• Themes: what questions or concepts recur across the literature?
• Debates, conflicts and contradictions: where do sources disagree?
• Pivotal publications: are there any influential theories or studies that changed the direction of the field?
• Gaps: what is missing from the literature? Are there weaknesses that need to be addressed?

This step will help you work out the structure of your literature review and (if applicable) show how your own research will contribute to existing knowledge.

• Most research has focused on young women.
• There is an increasing interest in the visual aspects of social media.
• But there is still a lack of robust research on highly visual platforms like Instagram and Snapchat—this is a gap that you could address in your own research.

There are various approaches to organizing the body of a literature review. Depending on the length of your literature review, you can combine several of these strategies (for example, your overall structure might be thematic, but each theme is discussed chronologically).

Chronological

The simplest approach is to trace the development of the topic over time. However, if you choose this strategy, be careful to avoid simply listing and summarizing sources in order.

Try to analyze patterns, turning points and key debates that have shaped the direction of the field. Give your interpretation of how and why certain developments occurred.

If you have found some recurring central themes, you can organize your literature review into subsections that address different aspects of the topic.

For example, if you are reviewing literature about inequalities in migrant health outcomes, key themes might include healthcare policy, language barriers, cultural attitudes, legal status, and economic access.

Methodological

If you draw your sources from different disciplines or fields that use a variety of research methods , you might want to compare the results and conclusions that emerge from different approaches. For example:

• Look at what results have emerged in qualitative versus quantitative research
• Discuss how the topic has been approached by empirical versus theoretical scholarship
• Divide the literature into sociological, historical, and cultural sources

Theoretical

A literature review is often the foundation for a theoretical framework . You can use it to discuss various theories, models, and definitions of key concepts.

You might argue for the relevance of a specific theoretical approach, or combine various theoretical concepts to create a framework for your research.

Like any other academic text , your literature review should have an introduction , a main body, and a conclusion . What you include in each depends on the objective of your literature review.

The introduction should clearly establish the focus and purpose of the literature review.

Depending on the length of your literature review, you might want to divide the body into subsections. You can use a subheading for each theme, time period, or methodological approach.

As you write, you can follow these tips:

• Summarize and synthesize: give an overview of the main points of each source and combine them into a coherent whole
• Analyze and interpret: don’t just paraphrase other researchers — add your own interpretations where possible, discussing the significance of findings in relation to the literature as a whole
• Critically evaluate: mention the strengths and weaknesses of your sources
• Write in well-structured paragraphs: use transition words and topic sentences to draw connections, comparisons and contrasts

In the conclusion, you should summarize the key findings you have taken from the literature and emphasize their significance.

When you’ve finished writing and revising your literature review, don’t forget to proofread thoroughly before submitting. Not a language expert? Check out Scribbr’s professional proofreading services !

This article has been adapted into lecture slides that you can use to teach your students about writing a literature review.

Scribbr slides are free to use, customize, and distribute for educational purposes.

Open Google Slides Download PowerPoint

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

• Sampling methods
• Simple random sampling
• Stratified sampling
• Cluster sampling
• Likert scales
• Reproducibility

 Statistics

• Null hypothesis
• Statistical power
• Probability distribution
• Effect size
• Poisson distribution

Research bias

• Optimism bias
• Cognitive bias
• Implicit bias
• Hawthorne effect
• Anchoring bias
• Explicit bias

A literature review is a survey of scholarly sources (such as books, journal articles, and theses) related to a specific topic or research question .

It is often written as part of a thesis, dissertation , or research paper , in order to situate your work in relation to existing knowledge.

There are several reasons to conduct a literature review at the beginning of a research project:

• To familiarize yourself with the current state of knowledge on your topic
• To ensure that you’re not just repeating what others have already done
• To identify gaps in knowledge and unresolved problems that your research can address
• To develop your theoretical framework and methodology
• To provide an overview of the key findings and debates on the topic

Writing the literature review shows your reader how your work relates to existing research and what new insights it will contribute.

The literature review usually comes near the beginning of your thesis or dissertation . After the introduction , it grounds your research in a scholarly field and leads directly to your theoretical framework or methodology .

A literature review is a survey of credible sources on a topic, often used in dissertations , theses, and research papers . Literature reviews give an overview of knowledge on a subject, helping you identify relevant theories and methods, as well as gaps in existing research. Literature reviews are set up similarly to other  academic texts , with an introduction , a main body, and a conclusion .

An  annotated bibliography is a list of  source references that has a short description (called an annotation ) for each of the sources. It is often assigned as part of the research process for a  paper .  

Cite this Scribbr article

If you want to cite this source, you can copy and paste the citation or click the “Cite this Scribbr article” button to automatically add the citation to our free Citation Generator.

McCombes, S. (2023, September 11). How to Write a Literature Review | Guide, Examples, & Templates. Scribbr. Retrieved April 15, 2024, from https://www.scribbr.com/dissertation/literature-review/

Is this article helpful?

Shona McCombes

Other students also liked, what is a theoretical framework | guide to organizing, what is a research methodology | steps & tips, how to write a research proposal | examples & templates, what is your plagiarism score.

Organizing Your Social Sciences Research Paper: Writing a Case Study

• Purpose of Guide
• Design Flaws to Avoid
• Independent and Dependent Variables
• Glossary of Research Terms
• Narrowing a Topic Idea
• Broadening a Topic Idea
• Extending the Timeliness of a Topic Idea
• Academic Writing Style
• Choosing a Title
• Making an Outline
• Paragraph Development
• Executive Summary
• The C.A.R.S. Model
• Background Information
• The Research Problem/Question
• Theoretical Framework
• Citation Tracking
• Content Alert Services
• Evaluating Sources
• Reading Research Effectively
• Primary Sources
• Secondary Sources
• Tiertiary Sources
• What Is Scholarly vs. Popular?
• Qualitative Methods
• Quantitative Methods
• Using Non-Textual Elements
• Limitations of the Study
• Common Grammar Mistakes
• Writing Concisely
• Avoiding Plagiarism
• Footnotes or Endnotes?
• Further Readings
• Annotated Bibliography
• Dealing with Nervousness
• Using Visual Aids
• Grading Someone Else's Paper
• Types of Structured Group Activities
• Group Project Survival Skills
• Multiple Book Review Essay
• Reviewing Collected Essays
• Writing a Case Study
• About Informed Consent
• Writing Field Notes
• Writing a Policy Memo
• Writing a Research Proposal
• Bibliography

The term case study refers to both a method of analysis and a specific research design for examining a problem, both of which are used in most circumstances to generalize across populations. This tab focuses on the latter--how to design and organize a research paper in the social sciences that analyzes a specific case.

A case study research paper examines a person, place, event, phenomenon, or other type of subject of analysis in order to extrapolate  key themes and results that help predict future trends, illuminate previously hidden issues that can be applied to practice, and/or provide a means for understanding an important research problem with greater clarity. A case study paper usually examines a single subject of analysis, but case study papers can also be designed as a comparative investigation that shows relationships between two or among more than two subjects. The methods used to study a case can rest within a quantitative, qualitative, or mixed-method investigative paradigm.

Case Studies . Writing@CSU. Colorado State University; Mills, Albert J. , Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research . Thousand Oaks, CA: SAGE Publications, 2010 ; “What is a Case Study?” In Swanborn, Peter G. Case Study Research: What, Why and How? London: SAGE, 2010.

How to Approach Writing a Case Study Research Paper

General information about how to choose a topic to investigate can be found under the " Choosing a Research Problem " tab in this writing guide. Review this page because it may help you identify a subject of analysis that can be investigated using a single case study design.

However, identifying a case to investigate involves more than choosing the research problem . A case study encompasses a problem contextualized around the application of in-depth analysis, interpretation, and discussion, often resulting in specific recommendations for action or for improving existing conditions. As Seawright and Gerring note, practical considerations such as time and access to information can influence case selection, but these issues should not be the sole factors used in describing the methodological justification for identifying a particular case to study. Given this, selecting a case includes considering the following:

• Does the case represent an unusual or atypical example of a research problem that requires more in-depth analysis? Cases often represent a topic that rests on the fringes of prior investigations because the case may provide new ways of understanding the research problem. For example, if the research problem is to identify strategies to improve policies that support girl's access to secondary education in predominantly Muslim nations, you could consider using Azerbaijan as a case study rather than selecting a more obvious nation in the Middle East. Doing so may reveal important new insights into recommending how governments in other predominantly Muslim nations can formulate policies that support improved access to education for girls.
• Does the case provide important insight or illuminate a previously hidden problem? In-depth analysis of a case can be based on the hypothesis that the case study will reveal trends or issues that have not been exposed in prior research or will reveal new and important implications for practice. For example, anecdotal evidence may suggest drug use among homeless veterans is related to their patterns of travel throughout the day. Assuming prior studies have not looked at individual travel choices as a way to study access to illicit drug use, a case study that observes a homeless veteran could reveal how issues of personal mobility choices facilitate regular access to illicit drugs. Note that it is important to conduct a thorough literature review to ensure that your assumption about the need to reveal new insights or previously hidden problems is valid and evidence-based.
• Does the case challenge and offer a counter-point to prevailing assumptions? Over time, research on any given topic can fall into a trap of developing assumptions based on outdated studies that are still applied to new or changing conditions or the idea that something should simply be accepted as "common sense," even though the issue has not been thoroughly tested in practice. A case may offer you an opportunity to gather evidence that challenges prevailing assumptions about a research problem and provide a new set of recommendations applied to practice that have not been tested previously. For example, perhaps there has been a long practice among scholars to apply a particular theory in explaining the relationship between two subjects of analysis. Your case could challenge this assumption by applying an innovative theoretical framework [perhaps borrowed from another discipline] to the study a case in order to explore whether this approach offers new ways of understanding the research problem. Taking a contrarian stance is one of the most important ways that new knowledge and understanding develops from existing literature.
• Does the case provide an opportunity to pursue action leading to the resolution of a problem? Another way to think about choosing a case to study is to consider how the results from investigating a particular case may result in findings that reveal ways in which to resolve an existing or emerging problem. For example, studying the case of an unforeseen incident, such as a fatal accident at a railroad crossing, can reveal hidden issues that could be applied to preventative measures that contribute to reducing the chance of accidents in the future. In this example, a case study investigating the accident could lead to a better understanding of where to strategically locate additional signals at other railroad crossings in order to better warn drivers of an approaching train, particularly when visibility is hindered by heavy rain, fog, or at night.
• Does the case offer a new direction in future research? A case study can be used as a tool for exploratory research that points to a need for further examination of the research problem. A case can be used when there are few studies that help predict an outcome or that establish a clear understanding about how best to proceed in addressing a problem. For example, after conducting a thorough literature review [very important!], you discover that little research exists showing the ways in which women contribute to promoting water conservation in rural communities of Uganda. A case study of how women contribute to saving water in a particular village can lay the foundation for understanding the need for more thorough research that documents how women in their roles as cooks and family caregivers think about water as a valuable resource within their community throughout rural regions of east Africa. The case could also point to the need for scholars to apply feminist theories of work and family to the issue of water conservation.

Eisenhardt, Kathleen M. “Building Theories from Case Study Research.” Academy of Management Review 14 (October 1989): 532-550; Emmel, Nick. Sampling and Choosing Cases in Qualitative Research: A Realist Approach . Thousand Oaks, CA: SAGE Publications, 2013; Gerring, John. “What Is a Case Study and What Is It Good for?” American Political Science Review 98 (May 2004): 341-354; Mills, Albert J. , Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research . Thousand Oaks, CA: SAGE Publications, 2010; Seawright, Jason and John Gerring. "Case Selection Techniques in Case Study Research." Political Research Quarterly 61 (June 2008): 294-308.

Structure and Writing Style

The purpose of a paper in the social sciences designed around a case study is to thoroughly investigate a subject of analysis in order to reveal a new understanding about the research problem and, in so doing, contributing new knowledge to what is already known from previous studies. In applied social sciences disciplines [e.g., education, social work, public administration, etc.], case studies may also be used to reveal best practices, highlight key programs, or investigate interesting aspects of professional work. In general, the structure of a case study research paper is not all that different from a standard college-level research paper. However, there are subtle differences you should be aware of. Here are the key elements to organizing and writing a case study research paper.

I.  Introduction

As with any research paper, your introduction should serve as a roadmap for your readers to ascertain the scope and purpose of your study . The introduction to a case study research paper, however, should not only describe the research problem and its significance, but you should also succinctly describe why the case is being used and how it relates to addressing the problem. The two elements should be linked. With this in mind, a good introduction answers these four questions:

• What was I studying? Describe the research problem and describe the subject of analysis you have chosen to address the problem. Explain how they are linked and what elements of the case will help to expand knowledge and understanding about the problem.
• Why was this topic important to investigate? Describe the significance of the research problem and state why a case study design and the subject of analysis that the paper is designed around is appropriate in addressing the problem.
• What did we know about this topic before I did this study? Provide background that helps lead the reader into the more in-depth literature review to follow. If applicable, summarize prior case study research applied to the research problem and why it fails to adequately address the research problem. Describe why your case will be useful. If no prior case studies have been used to address the research problem, explain why you have selected this subject of analysis.
• How will this study advance new knowledge or new ways of understanding? Explain why your case study will be suitable in helping to expand knowledge and understanding about the research problem.

Each of these questions should be addressed in no more than a few paragraphs. Exceptions to this can be when you are addressing a complex research problem or subject of analysis that requires more in-depth background information.

II.  Literature Review

The literature review for a case study research paper is generally structured the same as it is for any college-level research paper. The difference, however, is that the literature review is focused on providing background information and  enabling historical interpretation of the subject of analysis in relation to the research problem the case is intended to address . This includes synthesizing studies that help to:

• Place relevant works in the context of their contribution to understanding the case study being investigated . This would include summarizing studies that have used a similar subject of analysis to investigate the research problem. If there is literature using the same or a very similar case to study, you need to explain why duplicating past research is important [e.g., conditions have changed; prior studies were conducted long ago, etc.].
• Describe the relationship each work has to the others under consideration that informs the reader why this case is applicable . Your literature review should include a description of any works that support using the case to study the research problem and the underlying research questions.
• Identify new ways to interpret prior research using the case study . If applicable, review any research that has examined the research problem using a different research design. Explain how your case study design may reveal new knowledge or a new perspective or that can redirect research in an important new direction.
• Resolve conflicts amongst seemingly contradictory previous studies . This refers to synthesizing any literature that points to unresolved issues of concern about the research problem and describing how the subject of analysis that forms the case study can help resolve these existing contradictions.
• Point the way in fulfilling a need for additional research . Your review should examine any literature that lays a foundation for understanding why your case study design and the subject of analysis around which you have designed your study may reveal a new way of approaching the research problem or offer a perspective that points to the need for additional research.
• Expose any gaps that exist in the literature that the case study could help to fill . Summarize any literature that not only shows how your subject of analysis contributes to understanding the research problem, but how your case contributes to a new way of understanding the problem that prior research has failed to do.
• Locate your own research within the context of existing literature [very important!] . Collectively, your literature review should always place your case study within the larger domain of prior research about the problem. The overarching purpose of reviewing pertinent literature in a case study paper is to demonstrate that you have thoroughly identified and synthesized prior studies in the context of explaining the relevance of the case in addressing the research problem.

III.  Method

In this section, you explain why you selected a particular subject of analysis to study and the strategy you used to identify and ultimately decide that your case was appropriate in addressing the research problem. The way you describe the methods used varies depending on the type of subject of analysis that frames your case study.

If your subject of analysis is an incident or event . In the social and behavioral sciences, the event or incident that represents the case to be studied is usually bounded by time and place, with a clear beginning and end and with an identifiable location or position relative to its surroundings. The subject of analysis can be a rare or critical event or it can focus on a typical or regular event. The purpose of studying a rare event is to illuminate new ways of thinking about the broader research problem or to test a hypothesis. Critical incident case studies must describe the method by which you identified the event and explain the process by which you determined the validity of this case to inform broader perspectives about the research problem or to reveal new findings. However, the event does not have to be a rare or uniquely significant to support new thinking about the research problem or to challenge an existing hypothesis. For example, Walo, Bull, and Breen conducted a case study to identify and evaluate the direct and indirect economic benefits and costs of a local sports event in the City of Lismore, New South Wales, Australia. The purpose of their study was to provide new insights from measuring the impact of a typical local sports event that prior studies could not measure well because they focused on large "mega-events." Whether the event is rare or not, the methods section should include an explanation of the following characteristics of the event: a) when did it take place; b) what were the underlying circumstances leading to the event; c) what were the consequences of the event.

If your subject of analysis is a person. Explain why you selected this particular individual to be studied and describe what experience he or she has had that provides an opportunity to advance new understandings about the research problem. Mention any background about this person which might help the reader understand the significance of his/her experiences that make them worthy of study. This includes describing the relationships this person has had with other people, institutions, and/or events that support using him or her as the subject for a case study research paper. It is particularly important to differentiate the person as the subject of analysis from others and to succinctly explain how the person relates to examining the research problem.

If your subject of analysis is a place. In general, a case study that investigates a place suggests a subject of analysis that is unique or special in some way and that this uniqueness can be used to build new understanding or knowledge about the research problem. A case study of a place must not only describe its various attributes relevant to the research problem [e.g., physical, social, cultural, economic, political, etc.], but you must state the method by which you determined that this place will illuminate new understandings about the research problem. It is also important to articulate why a particular place as the case for study is being used if similar places also exist [i.e., if you are studying patterns of homeless encampments of veterans in open spaces, why study Echo Park in Los Angeles rather than Griffith Park?]. If applicable, describe what type of human activity involving this place makes it a good choice to study [e.g., prior research reveals Echo Park has more homeless veterans].

If your subject of analysis is a phenomenon. A phenomenon refers to a fact, occurrence, or circumstance that can be studied or observed but with the cause or explanation to be in question. In this sense, a phenomenon that forms your subject of analysis can encompass anything that can be observed or presumed to exist but is not fully understood. In the social and behavioral sciences, the case usually focuses on human interaction within a complex physical, social, economic, cultural, or political system. For example, the phenomenon could be the observation that many vehicles used by ISIS fighters are small trucks with English language advertisements on them. The research problem could be that ISIS fighters are difficult to combat because they are highly mobile. The research questions could be how and by what means are these vehicles used by ISIS being supplied to the militants and how might supply lines to these vehicles be cut? How might knowing the suppliers of these trucks from overseas reveal larger networks of collaborators and financial support? A case study of a phenomenon most often encompasses an in-depth analysis of a cause and effect that is grounded in an interactive relationship between people and their environment in some way.

NOTE:   The choice of the case or set of cases to study cannot appear random. Evidence that supports the method by which you identified and chose your subject of analysis should be linked to the findings from the literature review. Be sure to cite any prior studies that helped you determine that the case you chose was appropriate for investigating the research problem.

IV.  Discussion

The main elements of your discussion section are generally the same as any research paper, but centered around interpreting and drawing conclusions about the key findings from your case study. Note that a general social sciences research paper may contain a separate section to report findings. However, in a paper designed around a case study, it is more common to combine a description of the findings with the discussion about their implications. The objectives of your discussion section should include the following:

Reiterate the Research Problem/State the Major Findings Briefly reiterate the research problem you are investigating and explain why the subject of analysis around which you designed the case study were used. You should then describe the findings revealed from your study of the case using direct, declarative, and succinct proclamation of the study results. Highlight any findings that were unexpected or especially profound.

Explain the Meaning of the Findings and Why They are Important Systematically explain the meaning of your case study findings and why you believe they are important. Begin this part of the section by repeating what you consider to be your most important or surprising finding first, then systematically review each finding. Be sure to thoroughly extrapolate what your analysis of the case can tell the reader about situations or conditions beyond the actual case that was studied while, at the same time, being careful not to misconstrue or conflate a finding that undermines the external validity of your conclusions.

Relate the Findings to Similar Studies No study in the social sciences is so novel or possesses such a restricted focus that it has absolutely no relation to previously published research. The discussion section should relate your case study results to those found in other studies, particularly if questions raised from prior studies served as the motivation for choosing your subject of analysis. This is important because comparing and contrasting the findings of other studies helps to support the overall importance of your results and it highlights how and in what ways your case study design and the subject of analysis differs from prior research about the topic.

Consider Alternative Explanations of the Findings It is important to remember that the purpose of social science research is to discover and not to prove. When writing the discussion section, you should carefully consider all possible explanations for the case study results, rather than just those that fit your hypothesis or prior assumptions and biases. Be alert to what the in-depth analysis of the case may reveal about the research problem, including offering a contrarian perspective to what scholars have stated in prior research.

Acknowledge the Study's Limitations You can state the study's limitations in the conclusion section of your paper but describing the limitations of your subject of analysis in the discussion section provides an opportunity to identify the limitations and explain why they are not significant. This part of the discussion section should also note any unanswered questions or issues your case study could not address. More detailed information about how to document any limitations to your research can be found here .

Suggest Areas for Further Research Although your case study may offer important insights about the research problem, there are likely additional questions related to the problem that remain unanswered or findings that unexpectedly revealed themselves as a result of your in-depth analysis of the case. Be sure that the recommendations for further research are linked to the research problem and that you explain why your recommendations are valid in other contexts and based on the original assumptions of your study.

V.  Conclusion

As with any research paper, you should summarize your conclusion in clear, simple language; emphasize how the findings from your case study differs from or supports prior research and why. Do not simply reiterate the discussion section. Provide a synthesis of key findings presented in the paper to show how these converge to address the research problem. If you haven't already done so in the discussion section, be sure to document the limitations of your case study and needs for further research.

The function of your paper's conclusion is to: 1)  restate the main argument supported by the findings from the analysis of your case; 2) clearly state the context, background, and necessity of pursuing the research problem using a case study design in relation to an issue, controversy, or a gap found from reviewing the literature; and, 3) provide a place for you to persuasively and succinctly restate the significance of your research problem, given that the reader has now been presented with in-depth information about the topic.

Consider the following points to help ensure your conclusion is appropriate:

• If the argument or purpose of your paper is complex, you may need to summarize these points for your reader.
• If prior to your conclusion, you have not yet explained the significance of your findings or if you are proceeding inductively, use the conclusion of your paper to describe your main points and explain their significance.
• Move from a detailed to a general level of consideration of the case study's findings that returns the topic to the context provided by the introduction or within a new context that emerges from your case study findings.

Note that, depending on the discipline you are writing in and your professor's preferences, the concluding paragraph may contain your final reflections on the evidence presented applied to practice or on the essay's central research problem. However, the nature of being introspective about the subject of analysis you have investigated will depend on whether you are explicitly asked to express your observations in this way.

Problems to Avoid

Overgeneralization One of the goals of a case study is to lay a foundation for understanding broader trends and issues applied to similar circumstances. However, be careful when drawing conclusions from your case study. They must be evidence-based and grounded in the results of the study; otherwise, it is merely speculation. Looking at a prior example, it would be incorrect to state that a factor in improving girls access to education in Azerbaijan and the policy implications this may have for improving access in other Muslim nations is due to girls access to social media if there is no documentary evidence from your case study to indicate this. There may be anecdotal evidence that retention rates were better for girls who were on social media, but this observation would only point to the need for further research and would not be a definitive finding if this was not a part of your original research agenda.

Failure to Document Limitations No case is going to reveal all that needs to be understood about a research problem. Therefore, just as you have to clearly state the limitations of a general research study , you must describe the specific limitations inherent in the subject of analysis. For example, the case of studying how women conceptualize the need for water conservation in a village in Uganda could have limited application in other cultural contexts or in areas where fresh water from rivers or lakes is plentiful and, therefore, conservation is understood differently than preserving access to a scarce resource.

Failure to Extrapolate All Possible Implications Just as you don't want to over-generalize from your case study findings, you also have to be thorough in the consideration of all possible outcomes or recommendations derived from your findings. If you do not, your reader may question the validity of your analysis, particularly if you failed to document an obvious outcome from your case study research. For example, in the case of studying the accident at the railroad crossing to evaluate where and what types of warning signals should be located, you failed to take into consideration speed limit signage as well as warning signals. When designing your case study, be sure you have thoroughly addressed all aspects of the problem and do not leave gaps in your analysis.

Case Studies . Writing@CSU. Colorado State University; Gerring, John. Case Study Research: Principles and Practices . New York: Cambridge University Press, 2007; Merriam, Sharan B. Qualitative Research and Case Study Applications in Education . Rev. ed. San Francisco, CA: Jossey-Bass, 1998; Miller, Lisa L. “The Use of Case Studies in Law and Social Science Research.” Annual Review of Law and Social Science 14 (2018): TBD; Mills, Albert J., Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research . Thousand Oaks, CA: SAGE Publications, 2010; Putney, LeAnn Grogan. "Case Study." In Encyclopedia of Research Design , Neil J. Salkind, editor. (Thousand Oaks, CA: SAGE Publications, 2010), pp. 116-120; Simons, Helen. Case Study Research in Practice . London: SAGE Publications, 2009;  Kratochwill,  Thomas R. and Joel R. Levin, editors. Single-Case Research Design and Analysis: New Development for Psychology and Education .  Hilldsale, NJ: Lawrence Erlbaum Associates, 1992; Swanborn, Peter G. Case Study Research: What, Why and How? London : SAGE, 2010; Yin, Robert K. Case Study Research: Design and Methods . 6th edition. Los Angeles, CA, SAGE Publications, 2014; Walo, Maree, Adrian Bull, and Helen Breen. “Achieving Economic Benefits at Local Events: A Case Study of a Local Sports Event.” Festival Management and Event Tourism 4 (1996): 95-106.

Writing Tip

At Least Five Misconceptions about Case Study Research

Social science case studies are often perceived as limited in their ability to create new knowledge because they are not randomly selected and findings cannot be generalized to larger populations. Flyvbjerg examines five misunderstandings about case study research and systematically "corrects" each one. To quote, these are:

Misunderstanding 1 :  General, theoretical [context-independent knowledge is more valuable than concrete, practical (context-dependent) knowledge. Misunderstanding 2 :  One cannot generalize on the basis of an individual case; therefore, the case study cannot contribute to scientific development. Misunderstanding 3 :  The case study is most useful for generating hypotheses; that is, in the first stage of a total research process, whereas other methods are more suitable for hypotheses testing and theory building. Misunderstanding 4 :  The case study contains a bias toward verification, that is, a tendency to confirm the researcher’s preconceived notions. Misunderstanding 5 :  It is often difficult to summarize and develop general propositions and theories on the basis of specific case studies [p. 221].

While writing your paper, think introspectively about how you addressed these misconceptions because to do so can help you strengthen the validity and reliability of your research by clarifying issues of case selection, the testing and challenging of existing assumptions, the interpretation of key findings, and the summation of case outcomes. Think of a case study research paper as a complete, in-depth narrative about the specific properties and key characteristics of your subject of analysis applied to the research problem.

Flyvbjerg, Bent. “Five Misunderstandings About Case-Study Research.” Qualitative Inquiry 12 (April 2006): 219-245.

• << Previous: Reviewing Collected Essays
• Next: Writing a Field Report >>
• Last Updated: Jan 17, 2023 10:50 AM
• URL: https://libguides.pointloma.edu/ResearchPaper

Academia.edu no longer supports Internet Explorer.

To browse Academia.edu and the wider internet faster and more securely, please take a few seconds to  upgrade your browser .

Enter the email address you signed up with and we'll email you a reset link.

• We're Hiring!
• Help Center

A Review of the Literature on Case Study Research

This paper presents a review of the literature on case study research and comments on the ongoing debate of the value of case study. A research paradigm and its theoretical framework is described. This review focuses extensively on the positions of

Related Papers

Waleed Alabri

An Examination of Case Studies in Management Research: A Paradigmatic Bridge

Mehedi Masud

The paper maps the value of case study in management research. In particular, it deals with the paradigmatic aspects of case study as a research strategy. In order to analyse the convergence and divergence on different dimensions of the case study research, I focus on three well-known methodology experts, namely Robert Yin, Sharan Merriam and Robert Stake. I argue that case study is a comprehensive research strategy. It has the capacity to embrace paradigm plurality representing both inductive and deductive strategies. Because of its epistemological, ontological and methodological flexibility case study has become one of the established research approaches in management. There is no fixed set of methods for the case study research. This depends on the ontological presuppositions of the researchers. The significance of the ontology becomes apparent depending on the nature of the case and the types of the research questions. As case study research is reflexive, flexible and context-specific, it allows emerging contexts to shape methods. That is why it can act as a bridge across the research paradigms. I then look at the considerable influence that the case study approach has on the management research, i.e., the role for case study in the research process. Because of its overarching role, multi-paradigmatic approach can be adopted under case study research. Case study research is, in practice a varied methodology with paradigmatic pluralism covering an array of research methods and techniques and different levels of analysis.

International Leadership Journal

Michael Neubert

Extension of theory using a multiple-case study design can contribute value to a particular theoretical perspective and further define the boundaries of the original theory. Most organizations today operate in volatile economic and social environments. Qualitative research plays an essential role in the investigation of leadership and management problems, given that they remain complex social enigmas. The multiple case study design is a valuable qualitative research tool in studying the links between the personal, social, behavioral, psychological, organizational, cultural, and environmental factors that guide managerial and leadership development. Multiple-case studies can be used by both novice and experienced qualitative researchers to contribute original qualitative data to extant theory. Multiple-case study research is particularly suitable for responding to "how" and "why" and what Eisenhardt terms as "big picture" research questions that remain unanswered in the extant leadership and management literature.

Dhaulagiri Journal of Sociology and Anthropology

Thakur P R A S A D Bhatta

Case study research though increasingly popular in social sciences for positivist and intrepretivist research, a kind of confusion is prevalent when it is used ignoring its philosophical position. Arguably, the case study research is considered more appropriate for qualitative research because of its foremost strength ˗ the in-depth study of complex issues. This paper, drawing from the literature, discusses the philosophical position of case study research and argues that qualitative case study research is appropriate for theory building. For theory building, this paper follows the inductive approach guided by qualitative research paradigm and argues that it is not appropriate to assess theory building from the perspective of quantitative research. Very similar to other research methods, it is natural that the case study research has certain challenges; however, most of the challenges and misunderstandings overlap causing difficulty to understand the role of case study research. Hence, this paper aims to contribute to the understandings of the challenges and misunderstandings associated with the theory building from case study research. This paper argues that most of the challenges associated with theory building from case study can be addressed employing appropriate research strategies particularly clear understanding of philosophical stance and selection of appropriate case. The misunderstandings, on the other hand, are arisen due to the differences in the researcher's perspectives particularly positivistic thinking of them rather than the shortcomings inherent in the qualitative case study research design.

Florian Kohlbacher

This paper aims at exploring and discus­ sing the possibilities of applying qualitative content analysis as a (text) interpretation method in case study research. First, case study research as a research strategy within qualitative social research is briefly presented. Then, a basic introduction to (qualitative) content analysis as an interpretation method for qualitative interviews and other data material is given.

Assessment of Qualitative

15th NCVER conference

John Guenther

milton malaya

Lesley Bartlett

casestudies journal

Qualitative case-study research has experienced an upsurge in business management fields of inquiry in the recent past. A methodology is selection, justification and sequential arranging of activities, procedures and tasks in a research project. Research methodology can no longer be confined to a set of universally applicable rules, conventions and traditions. A research paradigm is a set of propositions that explains how the world is perceived. There are three basic paradigms: positivist, interpretive and critical. Qualitative " approaches to research " , " strategies of inquiry " and " varieties of methodologies " classified into five " types " or " traditions " namely; biography, phenomenology, grounded theory, ethnography and case study. The major criticism made of qualitative methods is that they are impressionistic and non-verifiable, post-positivists who reject this charge claiming that qualitative data is auditable and therefore dependable. The less structured qualitative methodologies reject many of the positivists " constructions over what constitutes rigour, favouring instead the flexibility, creativity and otherwise inaccessible insights afforded by alternative routes of inquiry that embrace storytelling, recollection, and dialogue. Case study research is not really a " methodology " or a method, rather an approach to research. Case studies can be ethnographic or not and some scholars identified it as a strategy of social inquiry. It is argued that, case studies are more appropriate to investigate causal relationships prevailing both in the business field as well as in wider society in general.

RELATED PAPERS

International journal of qualitative studies on health and well-being

Virginia Dickson-Swift

Patricia (Patsy) Duff

Complicity: An International Journal of Complexity Theory and Education

Lindsay Hetherington

saeed ahmed

Dr Jeff McCarthy

Molefe Coper Joseph

Carolina Chavez

Gloria Thakane Leutle

Alison While

Teresa Stone , Margaret McMillan

Liz Johnson

Nora Basurto , HOW Journal , Basurto Santos Nora M , Jolene Gregory

ZUHAL AHNAN

sosyalarastirmalar.com

Mansor Abu Talib

DR. HANNINGTONE GAYA

Jesus Manzano

Palliative Medicine

Carolyn Chew-graham

Qualitative Research Journal

Owen Barden

Organizational Research Methods

Cassie Earl

Raffaello Furlan

Sarah Quinton

Marlisa Abdul Rahim

Thabit Alomari

Journal of Agricultural Education

Public Management Review

Paul Hibbert

Angie Docherty

Heather Stewart , Rod Gapp

Jacqueline Fendt

Megan Peercy

Sadasfas Fsafs

Asma Humied

Sergio Ambrozio

Manual Therapy

Nikki Petty

Shafiullah Jan

WilliamG Tierney

Raymond Obeng

Felicity Kelliher

Jatin Thakur

•   We're Hiring!
•   Help Center
• Find new research papers in:
• Health Sciences
• Earth Sciences
• Cognitive Science
• Mathematics
• Computer Science
• Academia ©2024

Purdue Online Writing Lab Purdue OWL® College of Liberal Arts

Writing a Literature Review

Welcome to the Purdue OWL

This page is brought to you by the OWL at Purdue University. When printing this page, you must include the entire legal notice.

Copyright ©1995-2018 by The Writing Lab & The OWL at Purdue and Purdue University. All rights reserved. This material may not be published, reproduced, broadcast, rewritten, or redistributed without permission. Use of this site constitutes acceptance of our terms and conditions of fair use.

A literature review is a document or section of a document that collects key sources on a topic and discusses those sources in conversation with each other (also called synthesis ). The lit review is an important genre in many disciplines, not just literature (i.e., the study of works of literature such as novels and plays). When we say “literature review” or refer to “the literature,” we are talking about the research ( scholarship ) in a given field. You will often see the terms “the research,” “the scholarship,” and “the literature” used mostly interchangeably.

Where, when, and why would I write a lit review?

There are a number of different situations where you might write a literature review, each with slightly different expectations; different disciplines, too, have field-specific expectations for what a literature review is and does. For instance, in the humanities, authors might include more overt argumentation and interpretation of source material in their literature reviews, whereas in the sciences, authors are more likely to report study designs and results in their literature reviews; these differences reflect these disciplines’ purposes and conventions in scholarship. You should always look at examples from your own discipline and talk to professors or mentors in your field to be sure you understand your discipline’s conventions, for literature reviews as well as for any other genre.

A literature review can be a part of a research paper or scholarly article, usually falling after the introduction and before the research methods sections. In these cases, the lit review just needs to cover scholarship that is important to the issue you are writing about; sometimes it will also cover key sources that informed your research methodology.

Lit reviews can also be standalone pieces, either as assignments in a class or as publications. In a class, a lit review may be assigned to help students familiarize themselves with a topic and with scholarship in their field, get an idea of the other researchers working on the topic they’re interested in, find gaps in existing research in order to propose new projects, and/or develop a theoretical framework and methodology for later research. As a publication, a lit review usually is meant to help make other scholars’ lives easier by collecting and summarizing, synthesizing, and analyzing existing research on a topic. This can be especially helpful for students or scholars getting into a new research area, or for directing an entire community of scholars toward questions that have not yet been answered.

What are the parts of a lit review?

Most lit reviews use a basic introduction-body-conclusion structure; if your lit review is part of a larger paper, the introduction and conclusion pieces may be just a few sentences while you focus most of your attention on the body. If your lit review is a standalone piece, the introduction and conclusion take up more space and give you a place to discuss your goals, research methods, and conclusions separately from where you discuss the literature itself.

Introduction:

• An introductory paragraph that explains what your working topic and thesis is
• A forecast of key topics or texts that will appear in the review
• Potentially, a description of how you found sources and how you analyzed them for inclusion and discussion in the review (more often found in published, standalone literature reviews than in lit review sections in an article or research paper)
• Summarize and synthesize: Give an overview of the main points of each source and combine them into a coherent whole
• Analyze and interpret: Don’t just paraphrase other researchers – add your own interpretations where possible, discussing the significance of findings in relation to the literature as a whole
• Critically Evaluate: Mention the strengths and weaknesses of your sources
• Write in well-structured paragraphs: Use transition words and topic sentence to draw connections, comparisons, and contrasts.

Conclusion:

• Summarize the key findings you have taken from the literature and emphasize their significance
• Connect it back to your primary research question

How should I organize my lit review?

Lit reviews can take many different organizational patterns depending on what you are trying to accomplish with the review. Here are some examples:

• Chronological : The simplest approach is to trace the development of the topic over time, which helps familiarize the audience with the topic (for instance if you are introducing something that is not commonly known in your field). If you choose this strategy, be careful to avoid simply listing and summarizing sources in order. Try to analyze the patterns, turning points, and key debates that have shaped the direction of the field. Give your interpretation of how and why certain developments occurred (as mentioned previously, this may not be appropriate in your discipline — check with a teacher or mentor if you’re unsure).
• Thematic : If you have found some recurring central themes that you will continue working with throughout your piece, you can organize your literature review into subsections that address different aspects of the topic. For example, if you are reviewing literature about women and religion, key themes can include the role of women in churches and the religious attitude towards women.
• Qualitative versus quantitative research
• Empirical versus theoretical scholarship
• Divide the research by sociological, historical, or cultural sources
• Theoretical : In many humanities articles, the literature review is the foundation for the theoretical framework. You can use it to discuss various theories, models, and definitions of key concepts. You can argue for the relevance of a specific theoretical approach or combine various theorical concepts to create a framework for your research.

What are some strategies or tips I can use while writing my lit review?

Any lit review is only as good as the research it discusses; make sure your sources are well-chosen and your research is thorough. Don’t be afraid to do more research if you discover a new thread as you’re writing. More info on the research process is available in our "Conducting Research" resources .

As you’re doing your research, create an annotated bibliography ( see our page on the this type of document ). Much of the information used in an annotated bibliography can be used also in a literature review, so you’ll be not only partially drafting your lit review as you research, but also developing your sense of the larger conversation going on among scholars, professionals, and any other stakeholders in your topic.

Usually you will need to synthesize research rather than just summarizing it. This means drawing connections between sources to create a picture of the scholarly conversation on a topic over time. Many student writers struggle to synthesize because they feel they don’t have anything to add to the scholars they are citing; here are some strategies to help you:

• It often helps to remember that the point of these kinds of syntheses is to show your readers how you understand your research, to help them read the rest of your paper.
• Writing teachers often say synthesis is like hosting a dinner party: imagine all your sources are together in a room, discussing your topic. What are they saying to each other?
• Look at the in-text citations in each paragraph. Are you citing just one source for each paragraph? This usually indicates summary only. When you have multiple sources cited in a paragraph, you are more likely to be synthesizing them (not always, but often
• Read more about synthesis here.

The most interesting literature reviews are often written as arguments (again, as mentioned at the beginning of the page, this is discipline-specific and doesn’t work for all situations). Often, the literature review is where you can establish your research as filling a particular gap or as relevant in a particular way. You have some chance to do this in your introduction in an article, but the literature review section gives a more extended opportunity to establish the conversation in the way you would like your readers to see it. You can choose the intellectual lineage you would like to be part of and whose definitions matter most to your thinking (mostly humanities-specific, but this goes for sciences as well). In addressing these points, you argue for your place in the conversation, which tends to make the lit review more compelling than a simple reporting of other sources.

Harvey Cushing/John Hay Whitney Medical Library

• Collections
• Research Help

YSN Doctoral Programs: Steps in Conducting a Literature Review

• Biomedical Databases
• Global (Public Health) Databases
• Soc. Sci., History, and Law Databases
• Grey Literature
• Trials Registers
• Data and Statistics
• Public Policy
• Google Tips
• Recommended Books
• Steps in Conducting a Literature Review

What is a literature review?

A literature review is an integrated analysis -- not just a summary-- of scholarly writings and other relevant evidence related directly to your research question.  That is, it represents a synthesis of the evidence that provides background information on your topic and shows a association between the evidence and your research question.

A literature review may be a stand alone work or the introduction to a larger research paper, depending on the assignment.  Rely heavily on the guidelines your instructor has given you.

Why is it important?

A literature review is important because it:

• Explains the background of research on a topic.
• Demonstrates why a topic is significant to a subject area.
• Discovers relationships between research studies/ideas.
• Identifies major themes, concepts, and researchers on a topic.
• Identifies critical gaps and points of disagreement.
• Discusses further research questions that logically come out of the previous studies.

APA7 Style resources

APA Style Blog - for those harder to find answers

1. Choose a topic. Define your research question.

Your literature review should be guided by your central research question.  The literature represents background and research developments related to a specific research question, interpreted and analyzed by you in a synthesized way.

• Make sure your research question is not too broad or too narrow.  Is it manageable?
• Begin writing down terms that are related to your question. These will be useful for searches later.
• If you have the opportunity, discuss your topic with your professor and your class mates.

2. Decide on the scope of your review

How many studies do you need to look at? How comprehensive should it be? How many years should it cover? 

• This may depend on your assignment.  How many sources does the assignment require?

3. Select the databases you will use to conduct your searches.

Make a list of the databases you will search. 

Where to find databases:

• use the tabs on this guide
• Find other databases in the Nursing Information Resources web page
• More on the Medical Library web page
• ... and more on the Yale University Library web page

4. Conduct your searches to find the evidence. Keep track of your searches.

• Use the key words in your question, as well as synonyms for those words, as terms in your search. Use the database tutorials for help.
• Save the searches in the databases. This saves time when you want to redo, or modify, the searches. It is also helpful to use as a guide is the searches are not finding any useful results.
• Review the abstracts of research studies carefully. This will save you time.
• Use the bibliographies and references of research studies you find to locate others.
• Check with your professor, or a subject expert in the field, if you are missing any key works in the field.
• Ask your librarian for help at any time.
• Use a citation manager, such as EndNote as the repository for your citations. See the EndNote tutorials for help.

Review the literature

Some questions to help you analyze the research:

• What was the research question of the study you are reviewing? What were the authors trying to discover?
• Was the research funded by a source that could influence the findings?
• What were the research methodologies? Analyze its literature review, the samples and variables used, the results, and the conclusions.
• Does the research seem to be complete? Could it have been conducted more soundly? What further questions does it raise?
• If there are conflicting studies, why do you think that is?
• How are the authors viewed in the field? Has this study been cited? If so, how has it been analyzed?

Tips: 

• Review the abstracts carefully.  
• Keep careful notes so that you may track your thought processes during the research process.
• Create a matrix of the studies for easy analysis, and synthesis, across all of the studies.
• << Previous: Recommended Books
• Last Updated: Jan 4, 2024 10:52 AM
• URL: https://guides.library.yale.edu/YSNDoctoral

Organizing Your Social Sciences Research Assignments

• Annotated Bibliography
• Analyzing a Scholarly Journal Article
• Group Presentations
• Dealing with Nervousness
• Using Visual Aids
• Grading Someone Else's Paper
• Types of Structured Group Activities
• Group Project Survival Skills
• Leading a Class Discussion
• Multiple Book Review Essay
• Reviewing Collected Works
• Writing a Case Analysis Paper
• Writing a Case Study
• About Informed Consent
• Writing Field Notes
• Writing a Policy Memo
• Writing a Reflective Paper
• Writing a Research Proposal
• Generative AI and Writing
• Acknowledgments

A case study research paper examines a person, place, event, condition, phenomenon, or other type of subject of analysis in order to extrapolate  key themes and results that help predict future trends, illuminate previously hidden issues that can be applied to practice, and/or provide a means for understanding an important research problem with greater clarity. A case study research paper usually examines a single subject of analysis, but case study papers can also be designed as a comparative investigation that shows relationships between two or more subjects. The methods used to study a case can rest within a quantitative, qualitative, or mixed-method investigative paradigm.

Case Studies. Writing@CSU. Colorado State University; Mills, Albert J. , Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research . Thousand Oaks, CA: SAGE Publications, 2010 ; “What is a Case Study?” In Swanborn, Peter G. Case Study Research: What, Why and How? London: SAGE, 2010.

How to Approach Writing a Case Study Research Paper

General information about how to choose a topic to investigate can be found under the " Choosing a Research Problem " tab in the Organizing Your Social Sciences Research Paper writing guide. Review this page because it may help you identify a subject of analysis that can be investigated using a case study design.

However, identifying a case to investigate involves more than choosing the research problem . A case study encompasses a problem contextualized around the application of in-depth analysis, interpretation, and discussion, often resulting in specific recommendations for action or for improving existing conditions. As Seawright and Gerring note, practical considerations such as time and access to information can influence case selection, but these issues should not be the sole factors used in describing the methodological justification for identifying a particular case to study. Given this, selecting a case includes considering the following:

• The case represents an unusual or atypical example of a research problem that requires more in-depth analysis? Cases often represent a topic that rests on the fringes of prior investigations because the case may provide new ways of understanding the research problem. For example, if the research problem is to identify strategies to improve policies that support girl's access to secondary education in predominantly Muslim nations, you could consider using Azerbaijan as a case study rather than selecting a more obvious nation in the Middle East. Doing so may reveal important new insights into recommending how governments in other predominantly Muslim nations can formulate policies that support improved access to education for girls.
• The case provides important insight or illuminate a previously hidden problem? In-depth analysis of a case can be based on the hypothesis that the case study will reveal trends or issues that have not been exposed in prior research or will reveal new and important implications for practice. For example, anecdotal evidence may suggest drug use among homeless veterans is related to their patterns of travel throughout the day. Assuming prior studies have not looked at individual travel choices as a way to study access to illicit drug use, a case study that observes a homeless veteran could reveal how issues of personal mobility choices facilitate regular access to illicit drugs. Note that it is important to conduct a thorough literature review to ensure that your assumption about the need to reveal new insights or previously hidden problems is valid and evidence-based.
• The case challenges and offers a counter-point to prevailing assumptions? Over time, research on any given topic can fall into a trap of developing assumptions based on outdated studies that are still applied to new or changing conditions or the idea that something should simply be accepted as "common sense," even though the issue has not been thoroughly tested in current practice. A case study analysis may offer an opportunity to gather evidence that challenges prevailing assumptions about a research problem and provide a new set of recommendations applied to practice that have not been tested previously. For example, perhaps there has been a long practice among scholars to apply a particular theory in explaining the relationship between two subjects of analysis. Your case could challenge this assumption by applying an innovative theoretical framework [perhaps borrowed from another discipline] to explore whether this approach offers new ways of understanding the research problem. Taking a contrarian stance is one of the most important ways that new knowledge and understanding develops from existing literature.
• The case provides an opportunity to pursue action leading to the resolution of a problem? Another way to think about choosing a case to study is to consider how the results from investigating a particular case may result in findings that reveal ways in which to resolve an existing or emerging problem. For example, studying the case of an unforeseen incident, such as a fatal accident at a railroad crossing, can reveal hidden issues that could be applied to preventative measures that contribute to reducing the chance of accidents in the future. In this example, a case study investigating the accident could lead to a better understanding of where to strategically locate additional signals at other railroad crossings so as to better warn drivers of an approaching train, particularly when visibility is hindered by heavy rain, fog, or at night.
• The case offers a new direction in future research? A case study can be used as a tool for an exploratory investigation that highlights the need for further research about the problem. A case can be used when there are few studies that help predict an outcome or that establish a clear understanding about how best to proceed in addressing a problem. For example, after conducting a thorough literature review [very important!], you discover that little research exists showing the ways in which women contribute to promoting water conservation in rural communities of east central Africa. A case study of how women contribute to saving water in a rural village of Uganda can lay the foundation for understanding the need for more thorough research that documents how women in their roles as cooks and family caregivers think about water as a valuable resource within their community. This example of a case study could also point to the need for scholars to build new theoretical frameworks around the topic [e.g., applying feminist theories of work and family to the issue of water conservation].

Eisenhardt, Kathleen M. “Building Theories from Case Study Research.” Academy of Management Review 14 (October 1989): 532-550; Emmel, Nick. Sampling and Choosing Cases in Qualitative Research: A Realist Approach . Thousand Oaks, CA: SAGE Publications, 2013; Gerring, John. “What Is a Case Study and What Is It Good for?” American Political Science Review 98 (May 2004): 341-354; Mills, Albert J. , Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research . Thousand Oaks, CA: SAGE Publications, 2010; Seawright, Jason and John Gerring. "Case Selection Techniques in Case Study Research." Political Research Quarterly 61 (June 2008): 294-308.

Structure and Writing Style

The purpose of a paper in the social sciences designed around a case study is to thoroughly investigate a subject of analysis in order to reveal a new understanding about the research problem and, in so doing, contributing new knowledge to what is already known from previous studies. In applied social sciences disciplines [e.g., education, social work, public administration, etc.], case studies may also be used to reveal best practices, highlight key programs, or investigate interesting aspects of professional work.

In general, the structure of a case study research paper is not all that different from a standard college-level research paper. However, there are subtle differences you should be aware of. Here are the key elements to organizing and writing a case study research paper.

I.  Introduction

As with any research paper, your introduction should serve as a roadmap for your readers to ascertain the scope and purpose of your study . The introduction to a case study research paper, however, should not only describe the research problem and its significance, but you should also succinctly describe why the case is being used and how it relates to addressing the problem. The two elements should be linked. With this in mind, a good introduction answers these four questions:

• What is being studied? Describe the research problem and describe the subject of analysis [the case] you have chosen to address the problem. Explain how they are linked and what elements of the case will help to expand knowledge and understanding about the problem.
• Why is this topic important to investigate? Describe the significance of the research problem and state why a case study design and the subject of analysis that the paper is designed around is appropriate in addressing the problem.
• What did we know about this topic before I did this study? Provide background that helps lead the reader into the more in-depth literature review to follow. If applicable, summarize prior case study research applied to the research problem and why it fails to adequately address the problem. Describe why your case will be useful. If no prior case studies have been used to address the research problem, explain why you have selected this subject of analysis.
• How will this study advance new knowledge or new ways of understanding? Explain why your case study will be suitable in helping to expand knowledge and understanding about the research problem.

Each of these questions should be addressed in no more than a few paragraphs. Exceptions to this can be when you are addressing a complex research problem or subject of analysis that requires more in-depth background information.

II.  Literature Review

The literature review for a case study research paper is generally structured the same as it is for any college-level research paper. The difference, however, is that the literature review is focused on providing background information and  enabling historical interpretation of the subject of analysis in relation to the research problem the case is intended to address . This includes synthesizing studies that help to:

• Place relevant works in the context of their contribution to understanding the case study being investigated . This would involve summarizing studies that have used a similar subject of analysis to investigate the research problem. If there is literature using the same or a very similar case to study, you need to explain why duplicating past research is important [e.g., conditions have changed; prior studies were conducted long ago, etc.].
• Describe the relationship each work has to the others under consideration that informs the reader why this case is applicable . Your literature review should include a description of any works that support using the case to investigate the research problem and the underlying research questions.
• Identify new ways to interpret prior research using the case study . If applicable, review any research that has examined the research problem using a different research design. Explain how your use of a case study design may reveal new knowledge or a new perspective or that can redirect research in an important new direction.
• Resolve conflicts amongst seemingly contradictory previous studies . This refers to synthesizing any literature that points to unresolved issues of concern about the research problem and describing how the subject of analysis that forms the case study can help resolve these existing contradictions.
• Point the way in fulfilling a need for additional research . Your review should examine any literature that lays a foundation for understanding why your case study design and the subject of analysis around which you have designed your study may reveal a new way of approaching the research problem or offer a perspective that points to the need for additional research.
• Expose any gaps that exist in the literature that the case study could help to fill . Summarize any literature that not only shows how your subject of analysis contributes to understanding the research problem, but how your case contributes to a new way of understanding the problem that prior research has failed to do.
• Locate your own research within the context of existing literature [very important!] . Collectively, your literature review should always place your case study within the larger domain of prior research about the problem. The overarching purpose of reviewing pertinent literature in a case study paper is to demonstrate that you have thoroughly identified and synthesized prior studies in relation to explaining the relevance of the case in addressing the research problem.

III.  Method

In this section, you explain why you selected a particular case [i.e., subject of analysis] and the strategy you used to identify and ultimately decide that your case was appropriate in addressing the research problem. The way you describe the methods used varies depending on the type of subject of analysis that constitutes your case study.

If your subject of analysis is an incident or event . In the social and behavioral sciences, the event or incident that represents the case to be studied is usually bounded by time and place, with a clear beginning and end and with an identifiable location or position relative to its surroundings. The subject of analysis can be a rare or critical event or it can focus on a typical or regular event. The purpose of studying a rare event is to illuminate new ways of thinking about the broader research problem or to test a hypothesis. Critical incident case studies must describe the method by which you identified the event and explain the process by which you determined the validity of this case to inform broader perspectives about the research problem or to reveal new findings. However, the event does not have to be a rare or uniquely significant to support new thinking about the research problem or to challenge an existing hypothesis. For example, Walo, Bull, and Breen conducted a case study to identify and evaluate the direct and indirect economic benefits and costs of a local sports event in the City of Lismore, New South Wales, Australia. The purpose of their study was to provide new insights from measuring the impact of a typical local sports event that prior studies could not measure well because they focused on large "mega-events." Whether the event is rare or not, the methods section should include an explanation of the following characteristics of the event: a) when did it take place; b) what were the underlying circumstances leading to the event; and, c) what were the consequences of the event in relation to the research problem.

If your subject of analysis is a person. Explain why you selected this particular individual to be studied and describe what experiences they have had that provide an opportunity to advance new understandings about the research problem. Mention any background about this person which might help the reader understand the significance of their experiences that make them worthy of study. This includes describing the relationships this person has had with other people, institutions, and/or events that support using them as the subject for a case study research paper. It is particularly important to differentiate the person as the subject of analysis from others and to succinctly explain how the person relates to examining the research problem [e.g., why is one politician in a particular local election used to show an increase in voter turnout from any other candidate running in the election]. Note that these issues apply to a specific group of people used as a case study unit of analysis [e.g., a classroom of students].

If your subject of analysis is a place. In general, a case study that investigates a place suggests a subject of analysis that is unique or special in some way and that this uniqueness can be used to build new understanding or knowledge about the research problem. A case study of a place must not only describe its various attributes relevant to the research problem [e.g., physical, social, historical, cultural, economic, political], but you must state the method by which you determined that this place will illuminate new understandings about the research problem. It is also important to articulate why a particular place as the case for study is being used if similar places also exist [i.e., if you are studying patterns of homeless encampments of veterans in open spaces, explain why you are studying Echo Park in Los Angeles rather than Griffith Park?]. If applicable, describe what type of human activity involving this place makes it a good choice to study [e.g., prior research suggests Echo Park has more homeless veterans].

If your subject of analysis is a phenomenon. A phenomenon refers to a fact, occurrence, or circumstance that can be studied or observed but with the cause or explanation to be in question. In this sense, a phenomenon that forms your subject of analysis can encompass anything that can be observed or presumed to exist but is not fully understood. In the social and behavioral sciences, the case usually focuses on human interaction within a complex physical, social, economic, cultural, or political system. For example, the phenomenon could be the observation that many vehicles used by ISIS fighters are small trucks with English language advertisements on them. The research problem could be that ISIS fighters are difficult to combat because they are highly mobile. The research questions could be how and by what means are these vehicles used by ISIS being supplied to the militants and how might supply lines to these vehicles be cut off? How might knowing the suppliers of these trucks reveal larger networks of collaborators and financial support? A case study of a phenomenon most often encompasses an in-depth analysis of a cause and effect that is grounded in an interactive relationship between people and their environment in some way.

NOTE:   The choice of the case or set of cases to study cannot appear random. Evidence that supports the method by which you identified and chose your subject of analysis should clearly support investigation of the research problem and linked to key findings from your literature review. Be sure to cite any studies that helped you determine that the case you chose was appropriate for examining the problem.

IV.  Discussion

The main elements of your discussion section are generally the same as any research paper, but centered around interpreting and drawing conclusions about the key findings from your analysis of the case study. Note that a general social sciences research paper may contain a separate section to report findings. However, in a paper designed around a case study, it is common to combine a description of the results with the discussion about their implications. The objectives of your discussion section should include the following:

Reiterate the Research Problem/State the Major Findings Briefly reiterate the research problem you are investigating and explain why the subject of analysis around which you designed the case study were used. You should then describe the findings revealed from your study of the case using direct, declarative, and succinct proclamation of the study results. Highlight any findings that were unexpected or especially profound.

Explain the Meaning of the Findings and Why They are Important Systematically explain the meaning of your case study findings and why you believe they are important. Begin this part of the section by repeating what you consider to be your most important or surprising finding first, then systematically review each finding. Be sure to thoroughly extrapolate what your analysis of the case can tell the reader about situations or conditions beyond the actual case that was studied while, at the same time, being careful not to misconstrue or conflate a finding that undermines the external validity of your conclusions.

Relate the Findings to Similar Studies No study in the social sciences is so novel or possesses such a restricted focus that it has absolutely no relation to previously published research. The discussion section should relate your case study results to those found in other studies, particularly if questions raised from prior studies served as the motivation for choosing your subject of analysis. This is important because comparing and contrasting the findings of other studies helps support the overall importance of your results and it highlights how and in what ways your case study design and the subject of analysis differs from prior research about the topic.

Consider Alternative Explanations of the Findings Remember that the purpose of social science research is to discover and not to prove. When writing the discussion section, you should carefully consider all possible explanations revealed by the case study results, rather than just those that fit your hypothesis or prior assumptions and biases. Be alert to what the in-depth analysis of the case may reveal about the research problem, including offering a contrarian perspective to what scholars have stated in prior research if that is how the findings can be interpreted from your case.

Acknowledge the Study's Limitations You can state the study's limitations in the conclusion section of your paper but describing the limitations of your subject of analysis in the discussion section provides an opportunity to identify the limitations and explain why they are not significant. This part of the discussion section should also note any unanswered questions or issues your case study could not address. More detailed information about how to document any limitations to your research can be found here .

Suggest Areas for Further Research Although your case study may offer important insights about the research problem, there are likely additional questions related to the problem that remain unanswered or findings that unexpectedly revealed themselves as a result of your in-depth analysis of the case. Be sure that the recommendations for further research are linked to the research problem and that you explain why your recommendations are valid in other contexts and based on the original assumptions of your study.

V.  Conclusion

As with any research paper, you should summarize your conclusion in clear, simple language; emphasize how the findings from your case study differs from or supports prior research and why. Do not simply reiterate the discussion section. Provide a synthesis of key findings presented in the paper to show how these converge to address the research problem. If you haven't already done so in the discussion section, be sure to document the limitations of your case study and any need for further research.

The function of your paper's conclusion is to: 1) reiterate the main argument supported by the findings from your case study; 2) state clearly the context, background, and necessity of pursuing the research problem using a case study design in relation to an issue, controversy, or a gap found from reviewing the literature; and, 3) provide a place to persuasively and succinctly restate the significance of your research problem, given that the reader has now been presented with in-depth information about the topic.

Consider the following points to help ensure your conclusion is appropriate:

• If the argument or purpose of your paper is complex, you may need to summarize these points for your reader.
• If prior to your conclusion, you have not yet explained the significance of your findings or if you are proceeding inductively, use the conclusion of your paper to describe your main points and explain their significance.
• Move from a detailed to a general level of consideration of the case study's findings that returns the topic to the context provided by the introduction or within a new context that emerges from your case study findings.

Note that, depending on the discipline you are writing in or the preferences of your professor, the concluding paragraph may contain your final reflections on the evidence presented as it applies to practice or on the essay's central research problem. However, the nature of being introspective about the subject of analysis you have investigated will depend on whether you are explicitly asked to express your observations in this way.

Problems to Avoid

Overgeneralization One of the goals of a case study is to lay a foundation for understanding broader trends and issues applied to similar circumstances. However, be careful when drawing conclusions from your case study. They must be evidence-based and grounded in the results of the study; otherwise, it is merely speculation. Looking at a prior example, it would be incorrect to state that a factor in improving girls access to education in Azerbaijan and the policy implications this may have for improving access in other Muslim nations is due to girls access to social media if there is no documentary evidence from your case study to indicate this. There may be anecdotal evidence that retention rates were better for girls who were engaged with social media, but this observation would only point to the need for further research and would not be a definitive finding if this was not a part of your original research agenda.

Failure to Document Limitations No case is going to reveal all that needs to be understood about a research problem. Therefore, just as you have to clearly state the limitations of a general research study , you must describe the specific limitations inherent in the subject of analysis. For example, the case of studying how women conceptualize the need for water conservation in a village in Uganda could have limited application in other cultural contexts or in areas where fresh water from rivers or lakes is plentiful and, therefore, conservation is understood more in terms of managing access rather than preserving access to a scarce resource.

Failure to Extrapolate All Possible Implications Just as you don't want to over-generalize from your case study findings, you also have to be thorough in the consideration of all possible outcomes or recommendations derived from your findings. If you do not, your reader may question the validity of your analysis, particularly if you failed to document an obvious outcome from your case study research. For example, in the case of studying the accident at the railroad crossing to evaluate where and what types of warning signals should be located, you failed to take into consideration speed limit signage as well as warning signals. When designing your case study, be sure you have thoroughly addressed all aspects of the problem and do not leave gaps in your analysis that leave the reader questioning the results.

Case Studies. Writing@CSU. Colorado State University; Gerring, John. Case Study Research: Principles and Practices . New York: Cambridge University Press, 2007; Merriam, Sharan B. Qualitative Research and Case Study Applications in Education . Rev. ed. San Francisco, CA: Jossey-Bass, 1998; Miller, Lisa L. “The Use of Case Studies in Law and Social Science Research.” Annual Review of Law and Social Science 14 (2018): TBD; Mills, Albert J., Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research . Thousand Oaks, CA: SAGE Publications, 2010; Putney, LeAnn Grogan. "Case Study." In Encyclopedia of Research Design , Neil J. Salkind, editor. (Thousand Oaks, CA: SAGE Publications, 2010), pp. 116-120; Simons, Helen. Case Study Research in Practice . London: SAGE Publications, 2009;  Kratochwill,  Thomas R. and Joel R. Levin, editors. Single-Case Research Design and Analysis: New Development for Psychology and Education .  Hilldsale, NJ: Lawrence Erlbaum Associates, 1992; Swanborn, Peter G. Case Study Research: What, Why and How? London : SAGE, 2010; Yin, Robert K. Case Study Research: Design and Methods . 6th edition. Los Angeles, CA, SAGE Publications, 2014; Walo, Maree, Adrian Bull, and Helen Breen. “Achieving Economic Benefits at Local Events: A Case Study of a Local Sports Event.” Festival Management and Event Tourism 4 (1996): 95-106.

Writing Tip

At Least Five Misconceptions about Case Study Research

Social science case studies are often perceived as limited in their ability to create new knowledge because they are not randomly selected and findings cannot be generalized to larger populations. Flyvbjerg examines five misunderstandings about case study research and systematically "corrects" each one. To quote, these are:

Misunderstanding 1 :  General, theoretical [context-independent] knowledge is more valuable than concrete, practical [context-dependent] knowledge. Misunderstanding 2 :  One cannot generalize on the basis of an individual case; therefore, the case study cannot contribute to scientific development. Misunderstanding 3 :  The case study is most useful for generating hypotheses; that is, in the first stage of a total research process, whereas other methods are more suitable for hypotheses testing and theory building. Misunderstanding 4 :  The case study contains a bias toward verification, that is, a tendency to confirm the researcher’s preconceived notions. Misunderstanding 5 :  It is often difficult to summarize and develop general propositions and theories on the basis of specific case studies [p. 221].

While writing your paper, think introspectively about how you addressed these misconceptions because to do so can help you strengthen the validity and reliability of your research by clarifying issues of case selection, the testing and challenging of existing assumptions, the interpretation of key findings, and the summation of case outcomes. Think of a case study research paper as a complete, in-depth narrative about the specific properties and key characteristics of your subject of analysis applied to the research problem.

Flyvbjerg, Bent. “Five Misunderstandings About Case-Study Research.” Qualitative Inquiry 12 (April 2006): 219-245.

• << Previous: Writing a Case Analysis Paper
• Next: Writing a Field Report >>
• Last Updated: Mar 6, 2024 1:00 PM
• URL: https://libguides.usc.edu/writingguide/assignments

Advertisement

Toward a framework for selecting indicators of measuring sustainability and circular economy in the agri-food sector: a systematic literature review

• LIFE CYCLE SUSTAINABILITY ASSESSMENT
• Published: 02 March 2022

Cite this article

• Cecilia Silvestri   ORCID: orcid.org/0000-0003-2528-601X 1 ,
• Luca Silvestri   ORCID: orcid.org/0000-0002-6754-899X 2 ,
• Michela Piccarozzi   ORCID: orcid.org/0000-0001-9717-9462 1 &
• Alessandro Ruggieri 1  

2866 Accesses

11 Citations

9 Altmetric

Explore all metrics

A Correction to this article was published on 24 March 2022

This article has been updated

The implementation of sustainability and circular economy (CE) models in agri-food production can promote resource efficiency, reduce environmental burdens, and ensure improved and socially responsible systems. In this context, indicators for the measurement of sustainability play a crucial role. Indicators can measure CE strategies aimed to preserve functions, products, components, materials, or embodied energy. Although there is broad literature describing sustainability and CE indicators, no study offers such a comprehensive framework of indicators for measuring sustainability and CE in the agri-food sector.

Starting from this central research gap, a systematic literature review has been developed to measure the sustainability in the agri-food sector and, based on these findings, to understand how indicators are used and for which specific purposes.

The analysis of the results allowed us to classify the sample of articles in three main clusters (“Assessment-LCA,” “Best practice,” and “Decision-making”) and has shown increasing attention to the three pillars of sustainability (triple bottom line). In this context, an integrated approach of indicators (environmental, social, and economic) offers the best solution to ensure an easier transition to sustainability.

Conclusions

The sample analysis facilitated the identification of new categories of impact that deserve attention, such as the cooperation among stakeholders in the supply chain and eco-innovation.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price includes VAT (Russian Federation)

Instant access to the full article PDF.

Rent this article via DeepDyve

Institutional subscriptions

Source: Authors’ elaboration. Notes: The graph shows the temporal distribution of the articles under analysis

Source: Authors’ elaborations. Notes: The graph shows the time distribution of articles from the three major journals

Source: Authors’ elaboration. Notes: The graph shows the composition of the sample according to the three clusters identified by the analysis

Source: Authors’ elaboration. Notes: The graph shows the distribution of articles over time by cluster

Source: Authors’ elaboration. Notes: The graph shows the network visualization

Source: Authors’ elaboration. Notes: The graph shows the overlay visualization

Source: Authors’ elaboration. Notes: The graph shows the classification of articles by scientific field

Source: Authors’ elaboration. Notes: Article classification based on their cluster to which they belong and scientific field

Source: Authors’ elaboration

Source: Authors’ elaboration. Notes: The graph shows the distribution of items over time based on TBL

Source: Authors’ elaboration. Notes: The graph shows the Pareto diagram highlighting the most used indicators in literature for measuring sustainability in the agri-food sector

Source: Authors’ elaboration. Notes: The graph shows the distribution over time of articles divided into conceptual and empirical

Source: Authors’ elaboration. Notes: The graph shows the classification of articles, divided into conceptual and empirical, in-depth analysis

Source: Authors’ elaboration. Notes: The graph shows the geographical distribution of the authors

Source: Authors’ elaboration. Notes: The graph shows the distribution of authors according to the continent from which they originate

Source: Authors’ elaboration. Notes: The graph shows the time distribution of publication of authors according to the continent from which they originate

Source: Authors’ elaboration. Notes: Sustainability measurement indicators and impact categories of LCA, S-LCA, and LCC tools should be integrated in order to provide stakeholders with best practices as guidelines and tools to support both decision-making and measurement, according to the circular economy approach

Similar content being viewed by others

The socio-economic performance of agroecology. A review

Ioanna Mouratiadou, Alexander Wezel, … Paolo Bàrberi

The Circular Economy: An Interdisciplinary Exploration of the Concept and Application in a Global Context

Alan Murray, Keith Skene & Kathryn Haynes

Food systems and rural wellbeing: challenges and opportunities

Jim Woodhill, Avinash Kishore, … Saher Hasnain

Change history

24 march 2022.

A Correction to this paper has been published: https://doi.org/10.1007/s11367-022-02038-9

Acero AP, Rodriguez C, Ciroth A (2017) LCIA methods: impact assessment methods in life cycle assessment and their impact categories. Version 1.5.6. Green Delta 1–23

Accorsi R, Versari L, Manzini R (2015) Glass vs. plastic: Life cycle assessment of extra-virgin olive oil bottles across global supply chains. Sustain 7:2818–2840. https://doi.org/10.3390/su7032818

Adjei-Bamfo P, Maloreh-Nyamekye T, Ahenkan A (2019) The role of e-government in sustainable public procurement in developing countries: a systematic literature review. Resour Conserv Recycl 142:189–203. https://doi.org/10.1016/j.resconrec.2018.12.001

Article   Google Scholar  

Aivazidou E, Tsolakis N, Vlachos D, Iakovou E (2015) Water footprint management policies for agrifood supply chains: a critical taxonomy and a system dynamics modelling approach. Chem Eng Trans 43:115–120. https://doi.org/10.3303/CET1543020

Alhaddi H (2015) Triple bottom line and sustainability: a literature review. Bus Manag Stud 1:6–10

Allaoui H, Guo Y, Sarkis J (2019) Decision support for collaboration planning in sustainable supply chains. J Clean Prod 229:761–774. https://doi.org/10.1016/j.jclepro.2019.04.367

Alshqaqeeq F, Amin Esmaeili M, Overcash M, Twomey J (2020) Quantifying hospital services by carbon footprint: a systematic literature review of patient care alternatives. Resour Conserv Recycl 154:104560. https://doi.org/10.1016/j.resconrec.2019.104560

Anwar F, Chaudhry FN, Nazeer S et al (2016) Causes of ozone layer depletion and its effects on human: review. Atmos Clim Sci 06:129–134. https://doi.org/10.4236/acs.2016.61011

Aquilani B, Silvestri C, Ruggieri A (2016). A Systematic Literature Review on Total Quality Management Critical Success Factors and the Identification of New Avenues of Research. https://doi.org/10.1108/TQM-01-2016-0003

Aramyan L, Hoste R, Van Den Broek W et al (2011) Towards sustainable food production: a scenario study of the European pork sector. J Chain Netw Sci 11:177–189. https://doi.org/10.3920/JCNS2011.Qpork8

Arfini F, Antonioli F, Cozzi E et al (2019) Sustainability, innovation and rural development: the case of Parmigiano-Reggiano PDO. Sustain 11:1–17. https://doi.org/10.3390/su11184978

Assembly UG (2005) Resolution adopted by the general assembly. New York, NY

Avilés-Palacios C, Rodríguez-Olalla A (2021) The sustainability of waste management models in circular economies. Sustain 13:1–19. https://doi.org/10.3390/su13137105

Azevedo SG, Silva ME, Matias JCO, Dias GP (2018) The influence of collaboration initiatives on the sustainability of the cashew supply chain. Sustain 10:1–29. https://doi.org/10.3390/su10062075

Bajaj S, Garg R, Sethi M (2016) Total quality management: a critical literature review using Pareto analysis. Int J Product Perform Manag 67:128–154

Banasik A, Kanellopoulos A, Bloemhof-Ruwaard JM, Claassen GDH (2019) Accounting for uncertainty in eco-efficient agri-food supply chains: a case study for mushroom production planning. J Clean Prod 216:249–256. https://doi.org/10.1016/j.jclepro.2019.01.153

Barth H, Ulvenblad PO, Ulvenblad P (2017) Towards a conceptual framework of sustainable business model innovation in the agri-food sector: a systematic literature review. Sustain 9. https://doi.org/10.3390/su9091620

Bastas A, Liyanage K (2018) Sustainable supply chain quality management: a systematic review

Beckerman W (1992) Economic growth and the environment: whose growth? Whose environment? World Dev 20:481–496. https://doi.org/10.1016/0305-750X(92)90038-W

Belaud JP, Prioux N, Vialle C, Sablayrolles C (2019) Big data for agri-food 4.0: application to sustainability management for by-products supply chain. Comput Ind 111:41–50. https://doi.org/10.1016/j.compind.2019.06.006

Bele B, Norderhaug A, Sickel H (2018) Localized agri-food systems and biodiversity. Agric 8. https://doi.org/10.3390/agriculture8020022

Bilali H El, Calabrese G, Iannetta M et al (2020) Environmental sustainability of typical agro-food products: a scientifically sound and user friendly approach. New Medit 19:69–83. https://doi.org/10.30682/nm2002e

Blanc S, Massaglia S, Brun F et al (2019) Use of bio-based plastics in the fruit supply chain: an integrated approach to assess environmental, economic, and social sustainability. Sustain 11. https://doi.org/10.3390/su11092475

Bloemhof JM, van der Vorst JGAJ, Bastl M, Allaoui H (2015) Sustainability assessment of food chain logistics. Int J Logist Res Appl 18:101–117. https://doi.org/10.1080/13675567.2015.1015508

Bonisoli L, Galdeano-Gómez E, Piedra-Muñoz L (2018) Deconstructing criteria and assessment tools to build agri-sustainability indicators and support farmers’ decision-making process. J Clean Prod 182:1080–1094. https://doi.org/10.1016/j.jclepro.2018.02.055

Bonisoli L, Galdeano-Gómez E, Piedra-Muñoz L, Pérez-Mesa JC (2019) Benchmarking agri-food sustainability certifications: evidences from applying SAFA in the Ecuadorian banana agri-system. J Clean Prod 236. https://doi.org/10.1016/j.jclepro.2019.07.054

Bornmann L, Haunschild R, Hug SE (2018) Visualizing the context of citations referencing papers published by Eugene Garfield: a new type of keyword co-occurrence analysis. Scientometrics 114:427–437. https://doi.org/10.1007/s11192-017-2591-8

Boulding KE (1966) The economics of the coming spaceship earth. New York, 1-17

Bracquené E, Dewulf W, Duflou JR (2020) Measuring the performance of more circular complex product supply chains. Resour Conserv Recycl 154:104608. https://doi.org/10.1016/j.resconrec.2019.104608

Burck J, Hagen U, Bals C et al (2021) Climate Change Performance Index

Calisto Friant M, Vermeulen WJV, Salomone R (2020) A typology of circular economy discourses: navigating the diverse visions of a contested paradigm. Resour Conserv Recycl 161:104917. https://doi.org/10.1016/j.resconrec.2020.104917

Campbell BM, Beare DJ, Bennett EM et al (2017) Agriculture production as a major driver of the earth system exceeding planetary boundaries. Ecol Soc 22. https://doi.org/10.5751/ES-09595-220408

Capitanio F, Coppola A, Pascucci S (2010) Product and process innovation in the Italian food industry. Agribusiness 26:503–518. https://doi.org/10.1002/agr.20239

Caputo P, Zagarella F, Cusenza MA et al (2020) Energy-environmental assessment of the UIA-OpenAgri case study as urban regeneration project through agriculture. Sci Total Environ 729:138819. https://doi.org/10.1016/j.scitotenv.2020.138819

Article   CAS   Google Scholar  

Chabowski BR, Mena JA, Gonzalez-Padron TL (2011) The structure of sustainability research in marketing, 1958–2008: a basis for future research opportunities. J Acad Mark Sci 39:55–70. https://doi.org/10.1007/s11747-010-0212-7

Chadegani AA, Salehi H, Yunus M et al (2017) A comparison between two main academic literature collections : Web of Science and Scopus databases. Asian Soc Sci 9:18–26. https://doi.org/10.5539/ass.v9n5p18

Chams N, Guesmi B, Gil JM (2020) Beyond scientific contribution: assessment of the societal impact of research and innovation to build a sustainable agri-food sector. J Environ Manage 264. https://doi.org/10.1016/j.jenvman.2020.110455

Chandrakumar C, McLaren SJ, Jayamaha NP, Ramilan T (2019) Absolute sustainability-based life cycle assessment (ASLCA): a benchmarking approach to operate agri-food systems within the 2°C global carbon budget. J Ind Ecol 23:906–917. https://doi.org/10.1111/jiec.12830

Chaparro-Africano AM (2019) Toward generating sustainability indicators for agroecological markets. Agroecol Sustain Food Syst 43:40–66. https://doi.org/10.1080/21683565.2019.1566192

Colicchia C, Strozzi F (2012) Supply chain risk management: a new methodology for a systematic literature review

Conca L, Manta F, Morrone D, Toma P (2021) The impact of direct environmental, social, and governance reporting: empirical evidence in European-listed companies in the agri-food sector. Bus Strateg Environ 30:1080–1093. https://doi.org/10.1002/bse.2672

Coppola A, Ianuario S, Romano S, Viccaro M (2020) Corporate social responsibility in agri-food firms: the relationship between CSR actions and firm’s performance. AIMS Environ Sci 7:542–558. https://doi.org/10.3934/environsci.2020034

Corona B, Shen L, Reike D et al (2019) Towards sustainable development through the circular economy—a review and critical assessment on current circularity metrics. Resour Conserv Recycl 151:104498. https://doi.org/10.1016/j.resconrec.2019.104498

Correia MS (2019) Sustainability: An overview of the triple bottom line and sustainability implementation. Int J Strateg Eng 2:29–38.  https://doi.org/10.4018/IJoSE.2019010103

Coteur I, Marchand F, Debruyne L, Lauwers L (2019) Structuring the myriad of sustainability assessments in agri-food systems: a case in Flanders. J Clean Prod 209:472–480. https://doi.org/10.1016/j.jclepro.2018.10.066

CREA (2020) L’agricoltura italiana conta 2019

Crenna E, Sala S, Polce C, Collina E (2017) Pollinators in life cycle assessment: towards a framework for impact assessment. J Clean Prod 140:525–536. https://doi.org/10.1016/j.jclepro.2016.02.058

D’Eusanio M, Serreli M, Zamagni A, Petti L (2018) Assessment of social dimension of a jar of honey: a methodological outline. J Clean Prod 199:503–517. https://doi.org/10.1016/j.jclepro.2018.07.157

Dania WAP, Xing K, Amer Y (2018) Collaboration behavioural factors for sustainable agri-food supply chains: a systematic review. J Clean Prod 186:851–864

De Pascale A, Arbolino R, Szopik-Depczyńska K et al (2021) A systematic review for measuring circular economy: the 61 indicators. J Clean Prod 281. https://doi.org/10.1016/j.jclepro.2020.124942

De Schoenmakere M, Gillabel J (2017) Circular by design: products in the circular economy

Del Borghi A, Gallo M, Strazza C, Del Borghi M (2014) An evaluation of environmental sustainability in the food industry through life cycle assessment: the case study of tomato products supply chain. J Clean Prod 78:121–130. https://doi.org/10.1016/j.jclepro.2014.04.083

Del Borghi A, Strazza C, Magrassi F et al (2018) Life cycle assessment for eco-design of product–package systems in the food industry—the case of legumes. Sustain Prod Consum 13:24–36. https://doi.org/10.1016/j.spc.2017.11.001

Denyer D, Tranfield D (2009) Producing a systematic review. In: Buchanan B (ed) The sage handbook of organization research methods. Sage Publications Ltd, Cornwall, pp 671–689

Google Scholar  

Dietz T, Grabs J, Chong AE (2019) Mainstreamed voluntary sustainability standards and their effectiveness: evidence from the Honduran coffee sector. Regul Gov. https://doi.org/10.1111/rego.12239

Dixon-Woods M (2011) Using framework-based synthesis for conducting reviews of qualitative studies. BMC Med 9:9–10. https://doi.org/10.1186/1741-7015-9-39

do Canto NR, Bossle MB, Marques L, Dutra M, (2020) Supply chain collaboration for sustainability: a qualitative investigation of food supply chains in Brazil. Manag Environ Qual an Int J. https://doi.org/10.1108/MEQ-12-2019-0275

dos Santos RR, Guarnieri P (2020) Social gains for artisanal agroindustrial producers induced by cooperation and collaboration in agri-food supply chain. Soc Responsib J. https://doi.org/10.1108/SRJ-09-2019-0323

Doukidis GI, Matopoulos A, Vlachopoulou M, Manthou V, Manos B (2007) A conceptual framework for supply chain collaboration: empirical evidence from the agri‐food industry. Supply Chain Manag an Int Journal 12:177–186. https://doi.org/10.1108/13598540710742491

Durach CF, Kembro J, Wieland A (2017) A new paradigm for systematic literature reviews in supply chain management. J Supply Chain Manag 53:67–85. https://doi.org/10.1111/jscm.12145

Durán-Sánchez A, Álvarez-García J, Río-Rama D, De la Cruz M (2018) Sustainable water resources management: a bibliometric overview. Water 10:1–19. https://doi.org/10.3390/w10091191

Duru M, Therond O (2015) Livestock system sustainability and resilience in intensive production zones: which form of ecological modernization? Reg Environ Chang 15:1651–1665. https://doi.org/10.1007/s10113-014-0722-9

Edison Fondazione (2019) Le eccellenze agricole italiane. I primati europei e mondiali dell’Italia nei prodotti vegetali. Milan (IT)

Ehrenfeld JR (2005) The roots of sustainability. MIT Sloan Manag Rev 46(2)46:23–25

Elia V, Gnoni MG, Tornese F (2017) Measuring circular economy strategies through index methods: a critical analysis. J Clean Prod 142:2741–2751. https://doi.org/10.1016/j.jclepro.2016.10.196

Elkington J (1997) Cannibals with forks : the triple bottom line of 21st century business. Capstone, Oxford

Esposito B, Sessa MR, Sica D, Malandrino O (2020) Towards circular economy in the agri-food sector. A systematic literature review. Sustain 12. https://doi.org/10.3390/SU12187401

European Commission (2018) Agri-food trade in 2018

European Commission (2019) Monitoring EU agri-food trade: development until September 2019

Eurostat (2018) Small and large farms in the EU - statistics from the farm structure survey

FAO (2011) Biodiversity for food and agriculture. Italy, Rome

FAO (2012) Energy-smart food at FAO: an overview. Italy, Rome

FAO (2014) Food wastage footprint: fool cost-accounting

FAO (2016) The state of food and agriculture climate change, agriculture and food security. Italy, Rome

FAO (2017) The future of food and agriculture: trends and challenges. Italy, Rome

FAO (2020) The state of food security and nutrition in the world. Transforming Food Systems for Affordable Healthy Diets. Rome, Italy

Fassio F, Tecco N (2019) Circular economy for food: a systemic interpretation of 40 case histories in the food system in their relationships with SDGs. Systems 7:43. https://doi.org/10.3390/systems7030043

Fathollahi A, Coupe SJ (2021) Life cycle assessment (LCA) and life cycle costing (LCC) of road drainage systems for sustainability evaluation: quantifying the contribution of different life cycle phases. Sci Total Environ 776:145937. https://doi.org/10.1016/j.scitotenv.2021.145937

Ferreira VJ, Arnal ÁJ, Royo P et al (2019) Energy and resource efficiency of electroporation-assisted extraction as an emerging technology towards a sustainable bio-economy in the agri-food sector. J Clean Prod 233:1123–1132. https://doi.org/10.1016/j.jclepro.2019.06.030

Fiksel J (2006) A framework for sustainable remediation. JOM 8:15–22. https://doi.org/10.1021/es202595w

Flick U (2014) An introduction to qualitative research

Franciosi C, Voisin A, Miranda S et al (2020) Measuring maintenance impacts on sustainability of manufacturing industries : from a systematic literature review to a framework proposal. J Clean Prod 260:1–19. https://doi.org/10.1016/j.jclepro.2020.121065

Gaitán-Cremaschi D, Meuwissen MPM, Oude AGJML (2017) Total factor productivity: a framework for measuring agri-food supply chain performance towards sustainability. Appl Econ Perspect Policy 39:259–285. https://doi.org/10.1093/aepp/ppw008

Galdeano-Gómez E, Zepeda-Zepeda JA, Piedra-Muñoz L, Vega-López LL (2017) Family farm’s features influencing socio-economic sustainability: an analysis of the agri-food sector in southeast Spain. New Medit 16:50–61

Gallopín G, Herrero LMJ, Rocuts A (2014) Conceptual frameworks and visual interpretations of sustainability. Int J Sustain Dev 17:298–326. https://doi.org/10.1504/IJSD.2014.064183

Gallopín GC (2003) Sostenibilidad y desarrollo sostenible: un enfoque sistémico. Cepal, LATIN AMERICA

Garnett T (2013) Food sustainability: problems, perspectives and solutions. Proc Nutr Soc 72:29–39. https://doi.org/10.1017/S0029665112002947

Garofalo P, D’Andrea L, Tomaiuolo M et al (2017) Environmental sustainability of agri-food supply chains in Italy: the case of the whole-peeled tomato production under life cycle assessment methodology. J Food Eng 200:1–12. https://doi.org/10.1016/j.jfoodeng.2016.12.007

Gava O, Bartolini F, Venturi F et al (2018) A reflection of the use of the life cycle assessment tool for agri-food sustainability. Sustain 11. https://doi.org/10.3390/su11010071

Gazzola P, Querci E (2017) The connection between the quality of life and sustainable ecological development. Eur Sci J 7881:1857–7431

Geissdoerfer M, Savaget P, Bocken N, Hultink EJ (2017) The circular economy – a new sustainability paradigm ? The circular economy – a new sustainability paradigm ? J Clean Prod 143:757–768. https://doi.org/10.1016/j.jclepro.2016.12.048

Georgescu-Roegen N (1971) The entropy low and the economic process. Harward University Press, Cambridge Mass

Book   Google Scholar  

Gerbens-Leenes PW, Moll HC, Schoot Uiterkamp AJM (2003) Design and development of a measuring method for environmental sustainability in food production systems. Ecol Econ 46:231–248. https://doi.org/10.1016/S0921-8009(03)00140-X

Gésan-Guiziou G, Alaphilippe A, Aubin J et al (2020) Diversity and potentiality of multi-criteria decision analysis methods for agri-food research. Agron Sustain Dev 40. https://doi.org/10.1007/s13593-020-00650-3

Ghisellini P, Cialani C, Ulgiati S (2016) A review on circular economy: the expected transition to a balanced interplay of environmental and economic systems. J Clean Prod 114:11–32. https://doi.org/10.1016/j.jclepro.2015.09.007

Godoy-Durán Á, Galdeano- Gómez E, Pérez-Mesa JC, Piedra-Muñoz L (2017) Assessing eco-efficiency and the determinants of horticultural family-farming in southeast Spain. J Environ Manage 204:594–604. https://doi.org/10.1016/j.jenvman.2017.09.037

Gold S, Kunz N, Reiner G (2017) Sustainable global agrifood supply chains: exploring the barriers. J Ind Ecol 21:249–260. https://doi.org/10.1111/jiec.12440

Goucher L, Bruce R, Cameron DD et al (2017) The environmental impact of fertilizer embodied in a wheat-to-bread supply chain. Nat Plants 3:1–5. https://doi.org/10.1038/nplants.2017.12

Green A, Nemecek T, Chaudhary A, Mathys A (2020) Assessing nutritional, health, and environmental sustainability dimensions of agri-food production. Glob Food Sec 26:100406. https://doi.org/10.1016/j.gfs.2020.100406

Guinée JB, Heijungs R, Huppes G et al (2011) Life cycle assessment: past, present, and future †. Environ Sci Technol 45:90–96. https://doi.org/10.1021/es101316v

Guiomar N, Godinho S, Pinto-Correia T et al (2018) Typology and distribution of small farms in Europe: towards a better picture. Land Use Policy 75:784–798. https://doi.org/10.1016/j.landusepol.2018.04.012

Gunasekaran A, Patel C, McGaughey RE (2004) A framework for supply chain performance measurement. Int J Prod Econ 87:333–347. https://doi.org/10.1016/j.ijpe.2003.08.003

Gunasekaran A, Patel C, Tirtiroglu E (2001) Performance measures and metrics in a supply chain environment. Int J Oper Prod Manag 21:71–87. https://doi.org/10.1108/01443570110358468

Hamam M, Chinnici G, Di Vita G et al (2021) Circular economy models in agro-food systems: a review. Sustain 13

Harun SN, Hanafiah MM, Aziz NIHA (2021) An LCA-based environmental performance of rice production for developing a sustainable agri-food system in Malaysia. Environ Manage 67:146–161. https://doi.org/10.1007/s00267-020-01365-7

Harvey M, Pilgrim S (2011) The new competition for land: food, energy, and climate change. Food Policy 36:S40–S51. https://doi.org/10.1016/j.foodpol.2010.11.009

Hawkes C, Ruel MT (2006) Understanding the links between agriculture and health. DC: International Food Policy Research Institute. Washington, USA

Hellweg S, Milà i Canals L (2014) Emerging approaches, challenges and opportunities in life cycle assessment. Science (80)344:1109LP–1113. https://doi.org/10.1126/science.1248361

Higgins V, Dibden J, Cocklin C (2015) Private agri-food governance and greenhouse gas abatement: constructing a corporate carbon economy. Geoforum 66:75–84. https://doi.org/10.1016/j.geoforum.2015.09.012

Hill T (1995) Manufacturing strategy: text and cases., Macmillan

Hjeresen DD, Gonzales R (2020) Green chemistry promote sustainable agriculture?The rewards are higher yields and less environmental contamination. Environemental Sci Techonology 103–107

Horne R, Grant T, Verghese K (2009) Life cycle assessment: principles, practice, and prospects. Csiro Publishing, Collingwood, Australia

Horton P, Koh L, Guang VS (2016) An integrated theoretical framework to enhance resource efficiency, sustainability and human health in agri-food systems. J Clean Prod 120:164–169. https://doi.org/10.1016/j.jclepro.2015.08.092

Hospido A, Davis J, Berlin J, Sonesson U (2010) A review of methodological issues affecting LCA of novel food products. Int J Life Cycle Assess 15:44–52. https://doi.org/10.1007/s11367-009-0130-4

Huffman T, Liu J, Green M et al (2015) Improving and evaluating the soil cover indicator for agricultural land in Canada. Ecol Indic 48:272–281. https://doi.org/10.1016/j.ecolind.2014.07.008

Ilbery B, Maye D (2005) Food supply chains and sustainability: evidence from specialist food producers in the Scottish/English borders. Land Use Policy 22:331–344. https://doi.org/10.1016/j.landusepol.2004.06.002

Ingrao C, Faccilongo N, Valenti F et al (2019) Tomato puree in the Mediterranean region: an environmental life cycle assessment, based upon data surveyed at the supply chain level. J Clean Prod 233:292–313. https://doi.org/10.1016/j.jclepro.2019.06.056

Iocola I, Angevin F, Bockstaller C et al (2020) An actor-oriented multi-criteria assessment framework to support a transition towards sustainable agricultural systems based on crop diversification. Sustain 12. https://doi.org/10.3390/su12135434

Irabien A, Darton RC (2016) Energy–water–food nexus in the Spanish greenhouse tomato production. Clean Technol Environ Policy 18:1307–1316. https://doi.org/10.1007/s10098-015-1076-9

ISO 14040:2006 (2006) Environmental management — life cycle assessment — principles and framework

ISO 14044:2006 (2006) Environmental management — life cycle assessment — requirements and guidelines

ISO 15392:2008 (2008) Sustainability in building construction–general principles

Istat (2019) Andamento dell’economia agricola

Jaakkola E (2020) Designing conceptual articles : four approaches. AMS Rev 1–9. https://doi.org/10.1007/s13162-020-00161-0

Jin R, Yuan H, Chen Q (2019) Science mapping approach to assisting the review of construction and demolition waste management research published between 2009 and 2018. Resour Conserv Recycl 140:175–188. https://doi.org/10.1016/j.resconrec.2018.09.029

Johnston P, Everard M, Santillo D, Robèrt KH (2007) Reclaiming the definition of sustainability. Environ Sci Pollut Res Int 14:60–66. https://doi.org/10.1065/espr2007.01.375

Jorgensen SE, Burkhard B, Müller F (2013) Twenty volumes of ecological indicators-an accounting short review. Ecol Indic 28:4–9. https://doi.org/10.1016/j.ecolind.2012.12.018

Joshi S, Sharma M, Kler R (2020) Modeling circular economy dimensions in agri-tourism clusters: sustainable performance and future research directions. Int J Math Eng Manag Sci 5:1046–1061. https://doi.org/10.33889/IJMEMS.2020.5.6.080

Kamilaris A, Gao F, Prenafeta-Boldu FX, Ali MI (2017) Agri-IoT: a semantic framework for Internet of Things-enabled smart farming applications. In: 2016 IEEE 3rd World Forum on Internet of Things, WF-IoT 2016. pp 442–447

Karuppusami G, Gandhinathan R (2006) Pareto analysis of critical success factors of total quality management: a literature review and analysis. TQM Mag 18:372–385. https://doi.org/10.1108/09544780610671048

Kates RW, Parris TM, Leiserowitz AA (2005) What is sustainable development? Goals, indicators, values, and practice. Environ Sci Policy Sustain Dev 47:8–21. https://doi.org/10.1080/00139157.2005.10524444

Khounani Z, Hosseinzadeh-Bandbafha H, Moustakas K et al (2021) Environmental life cycle assessment of different biorefinery platforms valorizing olive wastes to biofuel, phosphate salts, natural antioxidant, and an oxygenated fuel additive (triacetin). J Clean Prod 278:123916. https://doi.org/10.1016/j.jclepro.2020.123916

Kitchenham B, Charters S (2007) Guidelines for performing systematic literature reviews in software engineering version 2.3. Engineering 45. https://doi.org/10.1145/1134285.1134500

Korhonen J, Nuur C, Feldmann A, Birkie SE (2018) Circular economy as an essentially contested concept. J Clean Prod 175:544–552. https://doi.org/10.1016/j.jclepro.2017.12.111

Kuisma M, Kahiluoto H (2017) Biotic resource loss beyond food waste: agriculture leaks worst. Resour Conserv Recycl 124:129–140. https://doi.org/10.1016/j.resconrec.2017.04.008

Laso J, Hoehn D, Margallo M et al (2018) Assessing energy and environmental efficiency of the Spanish agri-food system using the LCA/DEA methodology. Energies 11. https://doi.org/10.3390/en11123395

Lee KM (2007) So What is the “triple bottom line”? Int J Divers Organ Communities Nations Annu Rev 6:67–72. https://doi.org/10.18848/1447-9532/cgp/v06i06/39283

Lehmann RJ, Hermansen JE, Fritz M et al (2011) Information services for European pork chains - closing gaps in information infrastructures. Comput Electron Agric 79:125–136. https://doi.org/10.1016/j.compag.2011.09.002

León-Bravo V, Caniato F, Caridi M, Johnsen T (2017) Collaboration for sustainability in the food supply chain: a multi-stage study in Italy. Sustainability 9:1253

Lepage A (2009) The quality of life as attribute of sustainability. TQM J 21:105–115. https://doi.org/10.1108/17542730910938119

Li CZ, Zhao Y, Xiao B et al (2020) Research trend of the application of information technologies in construction and demolition waste management. J Clean Prod 263. https://doi.org/10.1016/j.jclepro.2020.121458

Lo Giudice A, Mbohwa C, Clasadonte MT, Ingrao C (2014) Life cycle assessment interpretation and improvement of the Sicilian artichokes production. Int J Environ Res 8:305–316. https://doi.org/10.22059/ijer.2014.721

Lueddeckens S, Saling P, Guenther E (2020) Temporal issues in life cycle assessment—a systematic review. Int J Life Cycle Assess 25:1385–1401. https://doi.org/10.1007/s11367-020-01757-1

Luo J, Ji C, Qiu C, Jia F (2018) Agri-food supply chain management: bibliometric and content analyses. Sustain 10. https://doi.org/10.3390/su10051573

Lynch J, Donnellan T, Finn JA et al (2019) Potential development of Irish agricultural sustainability indicators for current and future policy evaluation needs. J Environ Manage 230:434–445. https://doi.org/10.1016/j.jenvman.2018.09.070

MacArthur E (2013) Towards the circular economy. J Ind Ecol 2:23–44

MacArthur E (2017) Delivering the circular economy a toolkit for policymakers, The Ellen MacArthur Foundation

MacInnis DJ (2011) A framework for conceptual. J Mark 75:136–154. https://doi.org/10.1509/jmkg.75.4.136

Mangla SK, Luthra S, Rich N et al (2018) Enablers to implement sustainable initiatives in agri-food supply chains. Int J Prod Econ 203:379–393. https://doi.org/10.1016/j.ijpe.2018.07.012

Marotta G, Nazzaro C, Stanco M (2017) How the social responsibility creates value: models of innovation in Italian pasta industry. Int J Glob Small Bus 9:144–167. https://doi.org/10.1504/IJGSB.2017.088923

Martucci O, Arcese G, Montauti C, Acampora A (2019) Social aspects in the wine sector: comparison between social life cycle assessment and VIVA sustainable wine project indicators. Resources 8. https://doi.org/10.3390/resources8020069

Mayring P (2004) Forum : Qualitative social research Sozialforschung 2. History of content analysis. A Companion to Qual Res 1:159–176

McKelvey B (2002) Managing coevolutionary dynamics. In: 18th EGOS Conference. Barcelona, Spain, pp 1–21

McMichael AJ, Butler CD, Folke C (2003) New visions for addressing sustainability. Science (80- ) 302:1191–1920

Mehmood A, Ahmed S, Viza E et al (2021) Drivers and barriers towards circular economy in agri-food supply chain: a review. Bus Strateg Dev 1–17. https://doi.org/10.1002/bsd2.171

Mella P, Gazzola P (2011) Sustainability and quality of life: the development model. In: Kapounek S (ed) Enterprise and competitive environment. Mendel University: Brno, Czechia. 542–551

Merli R, Preziosi M, Acampora A (2018) How do scholars approach the circular economy ? A systematic literature review. J Clean Prod 178:703–722. https://doi.org/10.1016/j.jclepro.2017.12.112

Merli R, Preziosi M, Acampora A et al (2020) Recycled fibers in reinforced concrete: a systematic literature review. J Clean Prod 248:119207. https://doi.org/10.1016/j.jclepro.2019.119207

Miglietta PP, Morrone D (2018) Managing water sustainability: virtual water flows and economic water productivity assessment of the wine trade between Italy and the Balkans. Sustain 10. https://doi.org/10.3390/su10020543

Mitchell MGE, Chan KMA, Newlands NK, Ramankutty N (2020) Spatial correlations don’t predict changes in agricultural ecosystem services: a Canada-wide case study. Front Sustain Food Syst 4:1–17. https://doi.org/10.3389/fsufs.2020.539892

Moraga G, Huysveld S, Mathieux F et al (2019) Circular economy indicators: what do they measure?. Resour Conserv Recycl 146:452–461. https://doi.org/10.1016/j.resconrec.2019.03.045

Morrissey JE, Dunphy NP (2015) Towards sustainable agri-food systems: the role of integrated sustainability and value assessment across the supply-chain. Int J Soc Ecol Sustain Dev 6:41–58. https://doi.org/10.4018/IJSESD.2015070104

Moser G (2009) Quality of life and sustainability: toward person-environment congruity. J Environ Psychol 29:351–357. https://doi.org/10.1016/j.jenvp.2009.02.002

Muijs D (2010) Doing quantitative research in education with SPSS. London

Muller MF, Esmanioto F, Huber N, Loures ER (2019) A systematic literature review of interoperability in the green Building Information Modeling lifecycle. J Clean Prod 223:397–412. https://doi.org/10.1016/j.jclepro.2019.03.114

Muradin M, Joachimiak-Lechman K, Foltynowicz Z (2018) Evaluation of eco-efficiency of two alternative agricultural biogas plants. Appl Sci 8. https://doi.org/10.3390/app8112083

Naseer MA, ur R, Ashfaq M, Hassan S, et al (2019) Critical issues at the upstream level in sustainable supply chain management of agri-food industries: evidence from Pakistan’s citrus industry. Sustain 11:1–19. https://doi.org/10.3390/su11051326

Nattassha R, Handayati Y, Simatupang TM, Siallagan M (2020) Understanding circular economy implementation in the agri-food supply chain: the case of an Indonesian organic fertiliser producer. Agric Food Secur 9:1–16. https://doi.org/10.1186/s40066-020-00264-8

Nazari-Sharabian M, Ahmad S, Karakouzian M (2018) Climate change and eutrophication: a short review. Eng Technol Appl Sci Res 8:3668–3672. https://doi.org/10.5281/zenodo.2532694

Nazir N (2017) Understanding life cycle thinking and its practical application to agri-food system. Int J Adv Sci Eng Inf Technol 7:1861–1870. https://doi.org/10.18517/ijaseit.7.5.3578

Negra C, Remans R, Attwood S et al (2020) Sustainable agri-food investments require multi-sector co-development of decision tools. Ecol Indic 110:105851. https://doi.org/10.1016/j.ecolind.2019.105851

Newsham KK, Robinson SA (2009) Responses of plants in polar regions to UVB exposure: a meta-analysis. Glob Chang Biol 15:2574–2589. https://doi.org/10.1111/j.1365-2486.2009.01944.x

Niemeijer D, de Groot RS (2008) A conceptual framework for selecting environmental indicator sets. Ecol Indic 8:14–25. https://doi.org/10.1016/j.ecolind.2006.11.012

Niero M, Kalbar PP (2019) Coupling material circularity indicators and life cycle based indicators: a proposal to advance the assessment of circular economy strategies at the product level. Resour Conserv Recycl 140:305–312. https://doi.org/10.1016/j.resconrec.2018.10.002

Nikolaou IE, Tsagarakis KP (2021) An introduction to circular economy and sustainability: some existing lessons and future directions. Sustain Prod Consum 28:600–609. https://doi.org/10.1016/j.spc.2021.06.017

Notarnicola B, Hayashi K, Curran MA, Huisingh D (2012) Progress in working towards a more sustainable agri-food industry. J Clean Prod 28:1–8. https://doi.org/10.1016/j.jclepro.2012.02.007

Notarnicola B, Tassielli G, Renzulli PA, Monforti F (2017) Energy flows and greenhouses gases of EU (European Union) national breads using an LCA (life cycle assessment) approach. J Clean Prod 140:455–469. https://doi.org/10.1016/j.jclepro.2016.05.150

Opferkuch K, Caeiro S, Salomone R, Ramos TB (2021) Circular economy in corporate sustainability reporting: a review of organisational approaches. Bus Strateg Environ 1–22. https://doi.org/10.1002/bse.2854

Padilla-Rivera A, do Carmo BBT, Arcese G, Merveille N, (2021) Social circular economy indicators: selection through fuzzy delphi method. Sustain Prod Consum 26:101–110. https://doi.org/10.1016/j.spc.2020.09.015

Pagotto M, Halog A (2016) Towards a circular economy in Australian agri-food industry: an application of input-output oriented approaches for analyzing resource efficiency and competitiveness potential. J Ind Ecol 20:1176–1186. https://doi.org/10.1111/jiec.12373

Parent G, Lavallée S (2011) LCA potentials and limits within a sustainable agri-food statutory framework. Global food insecurity. Springer, Netherlands, Dordrecht, pp 161–171

Chapter   Google Scholar  

Pattey E, Qiu G (2012) Trends in primary particulate matter emissions from Canadian agriculture. J Air Waste Manag Assoc 62:737–747. https://doi.org/10.1080/10962247.2012.672058

Pauliuk S (2018) Critical appraisal of the circular economy standard BS 8001:2017 and a dashboard of quantitative system indicators for its implementation in organizations. Resour Conserv Recycl 129:81–92. https://doi.org/10.1016/j.resconrec.2017.10.019

Peano C, Migliorini P, Sottile F (2014) A methodology for the sustainability assessment of agri-food systems: an application to the slow food presidia project. Ecol Soc 19. https://doi.org/10.5751/ES-06972-190424

Peano C, Tecco N, Dansero E et al (2015) Evaluating the sustainability in complex agri-food systems: the SAEMETH framework. Sustain 7:6721–6741. https://doi.org/10.3390/su7066721

Pearce DW, Turner RK (1990) Economics of natural resources and the environment. Harvester Wheatsheaf, Hemel Hempstead, Herts

Pelletier N (2018) Social sustainability assessment of Canadian egg production facilities: methods, analysis, and recommendations. Sustain 10:1–17. https://doi.org/10.3390/su10051601

Peña C, Civit B, Gallego-Schmid A et al (2021) Using life cycle assessment to achieve a circular economy. Int J Life Cycle Assess 26:215–220. https://doi.org/10.1007/s11367-020-01856-z

Perez Neira D (2016) Energy sustainability of Ecuadorian cacao export and its contribution to climate change. A case study through product life cycle assessment. J Clean Prod 112:2560–2568. https://doi.org/10.1016/j.jclepro.2015.11.003

Pérez-Neira D, Grollmus-Venegas A (2018) Life-cycle energy assessment and carbon footprint of peri-urban horticulture. A comparative case study of local food systems in Spain. Landsc Urban Plan 172:60–68. https://doi.org/10.1016/j.landurbplan.2018.01.001

Pérez-Pons ME, Plaza-Hernández M, Alonso RS et al (2021) Increasing profitability and monitoring environmental performance: a case study in the agri-food industry through an edge-iot platform. Sustain 13:1–16. https://doi.org/10.3390/su13010283

Petti L, Serreli M, Di Cesare S (2018) Systematic literature review in social life cycle assessment. Int J Life Cycle Assess 23:422–431. https://doi.org/10.1007/s11367-016-1135-4

Pieroni MPP, McAloone TC, Pigosso DCA (2019) Business model innovation for circular economy and sustainability: a review of approaches. J Clean Prod 215:198–216. https://doi.org/10.1016/j.jclepro.2019.01.036

Polit DF, Beck CT (2004) Nursing research: principles and methods. Lippincott Williams & Wilkins, Philadelphia, PA

Porkka M, Gerten D, Schaphoff S et al (2016) Causes and trends of water scarcity in food production. Environ Res Lett 11:015001. https://doi.org/10.1088/1748-9326/11/1/015001

Prajapati H, Kant R, Shankar R (2019) Bequeath life to death: state-of-art review on reverse logistics. J Clean Prod 211:503–520. https://doi.org/10.1016/j.jclepro.2018.11.187

Priyadarshini P, Abhilash PC (2020) Policy recommendations for enabling transition towards sustainable agriculture in India. Land Use Policy 96:104718. https://doi.org/10.1016/j.landusepol.2020.104718

Pronti A, Coccia M (2020) Multicriteria analysis of the sustainability performance between agroecological and conventional coffee farms in the East Region of Minas Gerais (Brazil). Renew Agric Food Syst. https://doi.org/10.1017/S1742170520000332

Rabadán A, González-Moreno A, Sáez-Martínez FJ (2019) Improving firms’ performance and sustainability: the case of eco-innovation in the agri-food industry. Sustain 11. https://doi.org/10.3390/su11205590

Raut RD, Luthra S, Narkhede BE et al (2019) Examining the performance oriented indicators for implementing green management practices in the Indian agro sector. J Clean Prod 215:926–943. https://doi.org/10.1016/j.jclepro.2019.01.139

Recanati F, Marveggio D, Dotelli G (2018) From beans to bar: a life cycle assessment towards sustainable chocolate supply chain. Sci Total Environ 613–614:1013–1023. https://doi.org/10.1016/j.scitotenv.2017.09.187

Redclift M (2005) Sustainable development (1987–2005): an oxymoron comes of age. Sustain Dev 13:212–227. https://doi.org/10.1002/sd.281

Rezaei M, Soheilifard F, Keshvari A (2021) Impact of agrochemical emission models on the environmental assessment of paddy rice production using life cycle assessment approach. Energy Sources. Part A Recover Util Environ Eff 1–16

Rigamonti L, Mancini E (2021) Life cycle assessment and circularity indicators. Int J Life Cycle Assess. https://doi.org/10.1007/s11367-021-01966-2

Risku-Norja H, Mäenpää I (2007) MFA model to assess economic and environmental consequences of food production and consumption. Ecol Econ 60:700–711. https://doi.org/10.1016/j.ecolecon.2006.05.001

Ritzén S, Sandström GÖ (2017) Barriers to the circular economy – integration of perspectives and domains. Procedia CIRP 64:7–12. https://doi.org/10.1016/j.procir.2017.03.005

Rockström J, Steffen W, Noone K et al (2009) A safe operating space for humanity. Nature 461:472–475. https://doi.org/10.1038/461472a

Roos Lindgreen E, Mondello G, Salomone R et al (2021) Exploring the effectiveness of grey literature indicators and life cycle assessment in assessing circular economy at the micro level: a comparative analysis. Int J Life Cycle Assess. https://doi.org/10.1007/s11367-021-01972-4

Roselli L, Casieri A, De Gennaro BC et al (2020) Environmental and economic sustainability of table grape production in Italy. Sustain 12.  https://doi.org/10.3390/su12093670

Ross RB, Pandey V, Ross KL (2015) Sustainability and strategy in U.S. agri-food firms: an assessment of current practices. Int Food Agribus Manag Rev 18:17–48

Royo P, Ferreira VJ, López-Sabirón AM, Ferreira G. (2016) Hybrid diagnosis to characterise the energy and environmental enhancement of photovoltaic modules using smart materials. Energy 101:174–189. https://doi.org/10.1016/j.energy.2016.01.101

Ruggerio CA (2021) Sustainability and sustainable development: a review of principles and definitions. Sci Total Environ 786:147481. https://doi.org/10.1016/j.scitotenv.2021.147481

Ruiz-Almeida A, Rivera-Ferre MG (2019) Internationally-based indicators to measure agri-food systems sustainability using food sovereignty as a conceptual framework. Food Secur 11:1321–1337. https://doi.org/10.1007/s12571-019-00964-5

Ryan M, Hennessy T, Buckley C et al (2016) Developing farm-level sustainability indicators for Ireland using the Teagasc National Farm Survey. Irish J Agric Food Res 55:112–125. https://doi.org/10.1515/ijafr-2016-0011

Saade MRM, Yahia A, Amor B (2020) How has LCA been applied to 3D printing ? A systematic literature review and recommendations for future studies. J Clean Prod 244:118803. https://doi.org/10.1016/j.jclepro.2019.118803

Saitone TL, Sexton RJ (2017) Agri-food supply chain: evolution and performance with conflicting consumer and societal demands. Eur Rev Agric Econ 44:634–657. https://doi.org/10.1093/erae/jbx003

Salim N, Ab Rahman MN, Abd Wahab D (2019) A systematic literature review of internal capabilities for enhancing eco-innovation performance of manufacturing firms. J Clean Prod 209:1445–1460. https://doi.org/10.1016/j.jclepro.2018.11.105

Salimi N (2021) Circular economy in agri-food systems BT - strategic decision making for sustainable management of industrial networks. In: International S (ed) Rezaei J. Publishing, Cham, pp 57–70

Salomone R, Ioppolo G (2012) Environmental impacts of olive oil production: a life cycle assessment case study in the province of Messina (Sicily). J Clean Prod 28:88–100. https://doi.org/10.1016/j.jclepro.2011.10.004

Sánchez AD, Río DMDLC, García JÁ (2017) Bibliometric analysis of publications on wine tourism in the databases Scopus and WoS. Eur Res Manag Bus Econ 23:8–15. https://doi.org/10.1016/j.iedeen.2016.02.001

Saputri VHL, Sutopo W, Hisjam M, Ma’aram A (2019) Sustainable agri-food supply chain performance measurement model for GMO and non-GMO using data envelopment analysis method. Appl Sci 9. https://doi.org/10.3390/app9061199

Sassanelli C, Rosa P, Rocca R, Terzi S (2019) Circular economy performance assessment methods : a systematic literature review. J Clean Prod 229:440–453. https://doi.org/10.1016/j.jclepro.2019.05.019

Schiefer S, Gonzalez C, Flanigan S (2015) More than just a factor in transition processes? The role of collaboration in agriculture. In: Sutherland LA, Darnhofer I, Wilson GA, Zagata L (eds) Transition pathways towards sustainability in agriculture: case studies from Europe, CPI Group. Croydon, UK, pp. 83

Seuring S, Muller M (2008) From a literature review to a conceptual framework for sustainable supply chain management. J Clean Prod 16:1699–1710. https://doi.org/10.1016/j.jclepro.2008.04.020

Silvestri C, Silvestri L, Forcina A, et al (2021) Green chemistry contribution towards more equitable global sustainability and greater circular economy: A systematic literature review. J Clean Prod 294. https://doi.org/10.1016/j.jclepro.2021.126137

Smetana S, Schmitt E, Mathys A (2019) Sustainable use of Hermetia illucens insect biomass for feed and food: attributional and consequential life cycle assessment. Resour Conserv Recycl 144:285–296. https://doi.org/10.1016/j.resconrec.2019.01.042

Sonesson U, Berlin J, Ziegler F (2010) Environmental assessment and management in the food industry: life cycle assessment and related approaches. Woodhead Publishing, Cambridge

Soussana JF (2014) Research priorities for sustainable agri-food systems and life cycle assessment. J Clean Prod 73:19–23. https://doi.org/10.1016/j.jclepro.2014.02.061

Soylu A, Oruç C, Turkay M et al (2006) Synergy analysis of collaborative supply chain management in energy systems using multi-period MILP. Eur J Oper Res 174:387–403. https://doi.org/10.1016/j.ejor.2005.02.042

Spaiser V, Ranganathan S, Swain RB, Sumpter DJ (2017) The sustainable development oxymoron: quantifying and modelling the incompatibility of sustainable development goals. Int J Sustain Dev World Ecol 24:457–470. https://doi.org/10.1080/13504509.2016.1235624

Stewart R, Niero M (2018) Circular economy in corporate sustainability strategies: a review of corporate sustainability reports in the fast-moving consumer goods sector. Bus Strateg Environ 27:1005–1022. https://doi.org/10.1002/bse.2048

Stillitano T, Spada E, Iofrida N et al (2021) Sustainable agri-food processes and circular economy pathways in a life cycle perspective: state of the art of applicative research. Sustain 13:1–29. https://doi.org/10.3390/su13052472

Stone J, Rahimifard S (2018) Resilience in agri-food supply chains: a critical analysis of the literature and synthesis of a novel framework. Supply Chain Manag 23:207–238. https://doi.org/10.1108/SCM-06-2017-0201

Strazza C, Del Borghi A, Gallo M, Del Borghi M (2011) Resource productivity enhancement as means for promoting cleaner production: analysis of co-incineration in cement plants through a life cycle approach. J Clean Prod 19:1615–1621. https://doi.org/10.1016/j.jclepro.2011.05.014

Su B, Heshmati A, Geng Y, Yu X (2013) A review of the circular economy in China: moving from rhetoric to implementation. J Clean Prod 42:215–227. https://doi.org/10.1016/j.jclepro.2012.11.020

Suárez-Eiroa B, Fernández E, Méndez-Martínez G, Soto-Oñate D (2019) Operational principles of circular economy for sustainable development: linking theory and practice. J Clean Prod 214:952–961. https://doi.org/10.1016/j.jclepro.2018.12.271

Svensson G, Wagner B (2015) Implementing and managing economic, social and environmental efforts of business sustainability. Manag Environ Qual an Int Journal 26:195–213. https://doi.org/10.1108/MEQ-09-2013-0099

Tasca AL, Nessi S, Rigamonti L (2017) Environmental sustainability of agri-food supply chains: an LCA comparison between two alternative forms of production and distribution of endive in northern Italy. J Clean Prod 140:725–741. https://doi.org/10.1016/j.jclepro.2016.06.170

Tassielli G, Notarnicola B, Renzulli PA, Arcese G (2018) Environmental life cycle assessment of fresh and processed sweet cherries in southern Italy. J Clean Prod 171:184–197. https://doi.org/10.1016/j.jclepro.2017.09.227

Teixeira R, Pax S (2011) A survey of life cycle assessment practitioners with a focus on the agri-food sector. J Ind Ecol 15:817–820. https://doi.org/10.1111/j.1530-9290.2011.00421.x

Tobergte DR, Curtis S (2013) ILCD Handbook. J Chem Info Model. https://doi.org/10.278/33030

Tortorella MM, Di Leo S, Cosmi C et al (2020) A methodological integrated approach to analyse climate change effects in agri-food sector: the TIMES water-energy-food module. Int J Environ Res Public Health 17:1–21. https://doi.org/10.3390/ijerph17217703

Tranfield D, Denyer D, Smart P (2003) Towards a methodology for developing evidenceinformed management knowledge by means of systematic review. Br J Manag 14:207–222

Trivellas P, Malindretos G, Reklitis P (2020) Implications of green logistics management on sustainable business and supply chain performance: evidence from a survey in the greek agri-food sector. Sustain 12:1–29. https://doi.org/10.3390/su122410515

Tsangas M, Gavriel I, Doula M et al (2020) Life cycle analysis in the framework of agricultural strategic development planning in the Balkan region. Sustain 12:1–15. https://doi.org/10.3390/su12051813

Ülgen VS, Björklund M, Simm N (2019) Inter-organizational supply chain interaction for sustainability : a systematic literature review.

UNEP S (2020) Guidelines for social life cycle assessment of products and organizations 2020.

UNEP/SETAC (2009) United Nations Environment Programme-society of Environmental Toxicology and Chemistry. Guidelines for social life cycle assessment of products. France

United Nations (2011) Guiding principles on business and human rights. Implementing the United Nations “protect, respect and remedy” framework

United Nations (2015) Transforming our world: the 2030 agenda for sustainable development. sustainabledevelopment.un.org

Van Asselt ED, Van Bussel LGJ, Van Der Voet H et al (2014) A protocol for evaluating the sustainability of agri-food production systems - a case study on potato production in peri-urban agriculture in the Netherlands. Ecol Indic 43:315–321. https://doi.org/10.1016/j.ecolind.2014.02.027

Van der Ploeg JD (2014) Peasant-driven agricultural growth and food sovereignty. J Peasant Stud 41:999–1030. https://doi.org/10.1080/03066150.2013.876997

van Eck NJ, Waltman L (2010) Software survey: VOSviewer, a computer program for bibliometric mapping. Scientometrics 84:523–538. https://doi.org/10.1007/s11192-009-0146-3

Van Eck NJ, Waltman L (2019) Manual for VOSviwer version 1.6.10. CWTS Meaningful metrics 1–53

Vasa L, Angeloska A, Trendov NM (2017) Comparative analysis of circular agriculture development in selected Western Balkan countries based on sustainable performance indicators. Econ Ann 168:44–47. https://doi.org/10.21003/ea.V168-09

Verdecho MJ, Alarcón-Valero F, Pérez-Perales D et al (2020) A methodology to select suppliers to increase sustainability within supply chains. Cent Eur J Oper Res. https://doi.org/10.1007/s10100-019-00668-3

Vergine P, Salerno C, Libutti A et al (2017) Closing the water cycle in the agro-industrial sector by reusing treated wastewater for irrigation. J Clean Prod 164:587–596. https://doi.org/10.1016/j.jclepro.2017.06.239

WCED (1987) Our common future - call for action

Webster K (2013) What might we say about a circular economy? Some temptations to avoid if possible. World Futures 69:542–554

Wheaton E, Kulshreshtha S (2013) Agriculture and climate change: implications for environmental sustainability indicators. WIT Trans Ecol Environ 175:99–110. https://doi.org/10.2495/ECO130091

Wijewickrama MKCS, Chileshe N, Rameezdeen R, Ochoa JJ (2021) Information sharing in reverse logistics supply chain of demolition waste: a systematic literature review. J Clean Prod 280:124359. https://doi.org/10.1016/j.jclepro.2020.124359

Woodhouse A, Davis J, Pénicaud C, Östergren K (2018) Sustainability checklist in support of the design of food processing. Sustain Prod Consum 16:110–120. https://doi.org/10.1016/j.spc.2018.06.008

Wu R, Yang D, Chen J (2014) Social Life Cycle Assessment Revisited Sustain 6:4200–4226. https://doi.org/10.3390/su6074200

Yadav S, Luthra S, Garg D (2021) Modelling Internet of things (IoT)-driven global sustainability in multi-tier agri-food supply chain under natural epidemic outbreaks. Environ Sci Pollut Res 16633–16654. https://doi.org/10.1007/s11356-020-11676-1

Yee FM, Shaharudin MR, Ma G et al (2021) Green purchasing capabilities and practices towards Firm’s triple bottom line in Malaysia. J Clean Prod 307:127268. https://doi.org/10.1016/j.jclepro.2021.127268

Yigitcanlar T (2010) Rethinking sustainable development: urban management, engineering, and design. IGI Global

Zamagni A, Amerighi O, Buttol P (2011) Strengths or bias in social LCA? Int J Life Cycle Assess 16:596–598. https://doi.org/10.1007/s11367-011-0309-3

Download references

Author information

Authors and affiliations.

Department of Economy, Engineering, Society and Business Organization, University of “Tuscia, ” Via del Paradiso 47, 01100, Viterbo, VT, Italy

Cecilia Silvestri, Michela Piccarozzi & Alessandro Ruggieri

Department of Engineering, University of Rome “Niccolò Cusano, ” Via Don Carlo Gnocchi, 3, 00166, Rome, Italy

Luca Silvestri

You can also search for this author in PubMed   Google Scholar

Corresponding author

Correspondence to Cecilia Silvestri .

Ethics declarations

Competing interests.

The authors declare no competing interests.

Additional information

Communicated by Monia Niero

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

The original online version of this article was revised: a number of ill-placed paragraph headings were removed and the source indication "Authors' elaborations" was added to Tables 1-3.

Supplementary Information

Below is the link to the electronic supplementary material.

Supplementary file1 (DOCX 31 KB)

Rights and permissions.

Reprints and permissions

About this article

Silvestri, C., Silvestri, L., Piccarozzi, M. et al. Toward a framework for selecting indicators of measuring sustainability and circular economy in the agri-food sector: a systematic literature review. Int J Life Cycle Assess (2022). https://doi.org/10.1007/s11367-022-02032-1

Download citation

Received : 15 June 2021

Accepted : 16 February 2022

Published : 02 March 2022

DOI : https://doi.org/10.1007/s11367-022-02032-1

Share this article

Anyone you share the following link with will be able to read this content:

Sorry, a shareable link is not currently available for this article.

Provided by the Springer Nature SharedIt content-sharing initiative

• Agri-food sector
• Sustainability
• Circular economy
• Triple bottom line
• Life cycle assessment
• Find a journal
• Publish with us
• Track your research

• Open access
• Published: 14 October 2023

A scoping review of ‘Pacing’ for management of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS): lessons learned for the long COVID pandemic

• Nilihan E. M. Sanal-Hayes 1 , 7 ,
• Marie Mclaughlin 1 , 8 ,
• Lawrence D. Hayes 1 ,
• Jacqueline L. Mair   ORCID: orcid.org/0000-0002-1466-8680 2 , 3 ,
• Jane Ormerod 4 ,
• David Carless 1 ,
• Natalie Hilliard 5 ,
• Rachel Meach 1 ,
• Joanne Ingram 6 &
• Nicholas F. Sculthorpe 1  

Journal of Translational Medicine volume  21 , Article number:  720 ( 2023 ) Cite this article

3333 Accesses

5 Citations

21 Altmetric

Metrics details

Controversy over treatment for people with myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is a barrier to appropriate treatment. Energy management or pacing is a prominent coping strategy for people with ME/CFS. Whilst a definitive definition of pacing is not unanimous within the literature or healthcare providers, it typically comprises regulating activity to avoid post exertional malaise (PEM), the worsening of symptoms after an activity. Until now, characteristics of pacing, and the effects on patients’ symptoms had not been systematically reviewed. This is problematic as the most common approach to pacing, pacing prescription, and the pooled efficacy of pacing was unknown. Collating evidence may help advise those suffering with similar symptoms, including long COVID, as practitioners would be better informed on methodological approaches to adopt, pacing implementation, and expected outcomes.

In this scoping review of the literature, we aggregated type of, and outcomes of, pacing in people with ME/CFS.

Eligibility criteria

Original investigations concerning pacing were considered in participants with ME/CFS.

Sources of evidence

Six electronic databases (PubMed, Scholar, ScienceDirect, Scopus, Web of Science and the Cochrane Central Register of Controlled Trials [CENTRAL]) were searched; and websites MEPedia, Action for ME, and ME Action were also searched for grey literature, to fully capture patient surveys not published in academic journals.

A scoping review was conducted. Review selection and characterisation was performed by two independent reviewers using pretested forms.

Authors reviewed 177 titles and abstracts, resulting in 17 included studies: three randomised control trials (RCTs); one uncontrolled trial; one interventional case series; one retrospective observational study; two prospective observational studies; four cross-sectional observational studies; and five cross-sectional analytical studies. Studies included variable designs, durations, and outcome measures. In terms of pacing administration, studies used educational sessions and diaries for activity monitoring. Eleven studies reported benefits of pacing, four studies reported no effect, and two studies reported a detrimental effect in comparison to the control group.

Conclusions

Highly variable study designs and outcome measures, allied to poor to fair methodological quality resulted in heterogenous findings and highlights the requirement for more research examining pacing. Looking to the long COVID pandemic, our results suggest future studies should be RCTs utilising objectively quantified digitised pacing, over a longer duration of examination (i.e. longitudinal studies), using the core outcome set for patient reported outcome measures. Until these are completed, the literature base is insufficient to inform treatment practises for people with ME/CFS and long COVID.

Introduction

Post-viral illness occurs when individuals experience an extended period of feeling unwell after a viral infection [ 1 , 2 , 3 , 4 , 5 , 6 ]. While post-viral illness is generally a non-specific condition with a constellation of symptoms that may be experienced, fatigue is amongst the most commonly reported [ 7 , 8 , 9 ]. For example, our recent systematic review found there was up to 94% prevalence of fatigue in people following acute COVID-19 infection [ 3 ]. The increasing prevalence of long COVID has generated renewed interest in symptomology and time-course of post-viral fatigue, with PubMed reporting 72 articles related to “post-viral fatigue” between 2020 and 2022, but less than five for every year since 1990.

As the coronavirus pandemic developed, it became clear that a significant proportion of the population experienced symptoms which persisted beyond the initial viral infection, meeting the definition of a post-viral illness. Current estimates suggest one in eight people develop long COVID [ 10 ] and its symptomatology has repeatedly been suggested to overlap with clinical demonstrations of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS). In a study by Wong and Weitzer [ 11 ], long COVID symptoms from 21 studies were compared to a list of ME/CFS symptoms. Of the 29 known ME/CFS symptoms the authors reported that 25 (86%) were reported in at least one long COVID study suggesting significant similarities. Sukocheva et al. [ 12 ] reported that long COVID included changes in immune, cardiovascular, metabolic, gastrointestinal, nervous and autonomic systems. When observed from a pathological stance, this list of symptoms is shared with, or is similar to, the symptoms patients with ME/CFS describe [ 13 ]. In fact, a recent article reported 43% of people with long COVID are diagnosed with ME/CFS [ 13 ], evidencing the analogous symptom loads.

A striking commonality between long COVID and similar conditions such as ME/CFS is the worsening of symptoms including fatigue, pain, cognitive difficulties, sore throat, and/or swollen lymph nodes following exertion. Termed post exertional malaise (PEM) [ 14 , 15 , 16 , 17 ], lasting from hours to several days, it is arguably one of the most debilitating side effects experienced by those with ME/CFS [ 16 , 17 , 18 ]. PEM is associated with considerably reduced quality of life amongst those with ME/CFS, with reduced ability to perform activities of daily living, leading to restraints on social and family life, mental health comorbidities such as depression and anxiety, and devastating employment and financial consequences [ 19 , 20 , 21 , 22 ]. At present, there is no cure or pharmacological treatments for PEM, and therefore, effective symptom management strategies are required. This may be in part because the triggers of PEM are poorly understood, and there is little evidence for what causes PEM, beyond anecdotal evidence. The most common approach to manage PEM is to incorporate activity pacing into the day-to-day lives of those with ME/CFS with the intention of reducing the frequency of severity of bouts of PEM [ 23 ]. Pacing is defined as an approach where patients are encouraged to be as active as possible within the limits imposed by the illness [ 23 , 24 , 25 ]. In practice, pacing requires individuals to determine a level at which they can function, but which does not lead to a marked increase in fatigue and other symptoms [ 26 , 27 ].

Although long COVID is a new condition [ 3 , 14 ], the available evidence suggests substantial overlap with the symptoms of conditions such as ME/CFS and it is therefore pragmatic to consider the utility of management strategies (such as pacing) used in ME/CFS for people with long COVID. In fact, a recent Delphi study recommended that management of long COVID should incorporate careful pacing to avoid PEM relapse [ 28 ]. This position was enforced by a multidisciplinary consensus statement considering treatment of fatigue in long COVID, recommending energy conservation strategies (including pacing) for people with long COVID [ 29 ]. Given the estimated > 2 million individuals who have experienced long COVID in the UK alone [ 30 , 31 , 32 ], there is an urgent need for evidence-based public health strategies. In this context, it seems pragmatic to borrow from the ME/CFS literature.

From a historical perspective, the 2007 NICE guidelines for people with ME/CFS advised both cognitive behavioural therapy (CBT) and graded exercise therapy (GET) should be offered to people with ME/CFS [ 33 ]. As of the 2021 update, NICE guidelines for people with ME/CFS do not advise CBT or GET, and the only recommended management strategy is pacing [ 34 ]. In the years between changes to these guidelines, the landmark PACE trial [ 35 ] was published in 2011. This large, randomised control trial (RCT; n = 639) compared pacing with CBT and reported GET and CBT were more effective than pacing for improving symptoms. Yet, this study has come under considerable criticism from patient groups and clinicians alike [ 36 , 37 , 38 , 39 ]. This may partly explain why NICE do not advise CBT or GET as of 2021, and only recommend pacing for symptom management people with ME/CFS [ 34 ]. There has been some controversy over best treatment for people with ME/CFS in the literature and support groups, potentially amplified by the ambiguity of evidence for pacing efficacy and how pacing should be implemented. As such, before pacing can be advised for people with long COVID, it is imperative previous literature concerning pacing is systematically reviewed. This is because a consensus is needed within the literature for implementing pacing so practitioners treating people with ME/CFS or long COVID can do so effectively. A lack of agreement in pacing implementation is a barrier to adoption for both practitioners and patients. Despite several systematic reviews concerning pharmacological interventions or cognitive behavioural therapy in people with ME/CFS [ 36 , 40 , 41 ], to date, there are no systematic reviews concerning pacing.

Despite the widespread use of pacing, the literature base is limited and includes clinical commentaries, case studies, case series, and few randomised control trials. Consequently, while a comprehensive review of the effects of pacing in ME/CFS is an essential tool to guide symptom management advice, the available literature means that effective pooling of data is not feasible [ 42 ] and therefore, a traditional systematic review and meta-analysis, with a tightly focussed research question would be premature [ 43 ]. Consequently, we elected to undertake a scoping review. This approach retains the systematic approach to literature searching but aims to map out the current state of the research [ 43 ]. Using the framework of Arksey and O'Malley [ 44 ], a scoping review aims to use a broad set of search terms and include a wide range of study designs and methods (in contrast to a systematic review [ 44 ]). This approach, has the benefit of clarifying key concepts, surveying current data collection approaches, and identifying critical knowledge gaps.

We aimed to provide an overview of existing literature concerning pacing in ME/CFS. Our three specific objectives of this scoping review were to (1) conduct a systematic search of the published literature concerning ME/CFS and pacing, (2) map characteristics and methodologies used, and (3) provide recommendations for the advancement of the research area.

Protocol and registration

The review was conducted and reported according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for scoping reviews (PRISMA-ScR) guidelines [ 45 ] and the five-stage framework outlined in Arksey and O’Malley [ 44 ]. Registration is not recommended for scoping reviews.

Studies that met the following criteria were included in this review: (1) published as a full-text manuscript; (2) not a review; (3) participants with ME/CFS; (4) studies employed a pacing intervention or retrospective analysis of pacing or a case study of pacing. Studies utilising sub-analysis of the pacing, graded activity, and cognitive behaviour therapy: a randomised evaluation (PACE) trial were included as these have different outcome measures and, as this is not a meta-analysis, this will not influence effect size estimates. Additionally, due to the paucity of evidence, grey literature has also been included in this review.

Search strategy

The search strategy consisted of a combination of free-text and MeSH terms relating to ME/CFS and pacing, which were developed through an examination of published original literature and review articles. Example search terms for PubMed included: ‘ME/CFS’ OR ‘ME’ OR ‘CFS’ OR ‘chronic fatigue syndrome’ OR ‘PEM’ OR ‘post exertional malaise’ OR ‘pene’ OR ‘post-exertion neurogenic exhaust’ AND ‘pacing’ OR ‘adaptive pacing’. The search was performed within title/abstract. Full search terms can be found in Additional file 1 .

Information sources

Six electronic databases [PubMed, Scholar, ScienceDirect, Scopus, Web of Science, and the Cochrane Central Register of Controlled Trials (CENTRAL)] were searched to identify original research articles published from the earliest available date up until 02/02/2022. Additional records were identified through reference lists of included studies. ‘Grey literature’ repositories including MEPedia, Action for ME, and ME Action were also searched with the same terms.

Study selection and data items

Once each database search was completed and manuscripts were sourced, all studies were downloaded into a single reference list (Zotero, version 6.0.23) and duplicates were removed. Titles and abstracts were screened for eligibility by two reviewers independently and discrepancies were resolved through discussion between reviewers. Subsequently, full text papers of potentially relevant studies were retrieved and assessed for eligibility by the same two reviewers independently. Any uncertainty by reviewers was discussed in consensus meetings and resolved by agreement. Data extracted from each study included sample size, participant characteristics, study design, trial registration details, study location, pacing description (type), intervention duration, intervention adherence, outcome variables, and main outcome data. Descriptions were extracted with as much detail as was provided by the authors. Study quality was assessed using the Physiotherapy Evidence Database (PEDro) scale [ 46 , 47 ].

Role of the funding source

The study sponsors had no role in study design, data collection, analysis, or interpretation, nor writing the report, nor submitting the paper for publication.

Study selection

After the initial database search, 281 records were identified (see Fig.  1 ). Once duplicates were removed, 177 titles and abstracts were screened for inclusion resulting in 22 studies being retrieved as full text and assessed for eligibility. Of those, five were excluded, and 17 articles remained and were used in the final qualitative synthesis.

Schematic flow diagram describing exclusions of potential studies and final number of studies. RCT = randomized control trial. CT = controlled trial. UCT = uncontrolled trial

Study characteristics

Study characteristics are summarised in Table 1 . Of the 17 studies included, three were randomised control trials (RCTs [ 35 , 48 , 49 ]); one was an uncontrolled trial [ 50 ]; one was a case series [ 51 ]; one was a retrospective observational study [ 52 ], two were prospective observational studies [ 53 , 54 ]; four were cross-sectional observational studies [ 25 , 55 , 56 ]; and five were cross-sectional analytical studies [ 57 , 58 , 59 , 60 , 61 ] including sub-analysis of the PACE trial [ 35 , 56 , 59 , 61 ]. Seven of the studies were registered trials [ 35 , 48 , 49 , 50 , 56 , 57 , 58 ]. Diagnostic criteria for ME/CFS are summarised in Table 2 .

Types of pacing

Pacing interventions.

Of the 17 studies included, five implemented their own pacing interventions and will be discussed in this section. Sample sizes ranged from n = 7 in an interventional case series [ 51 ] to n = 641 participants in the largest RCT [ 35 ]. The first of these five studies considered an education session on pacing and self-management as the ‘pacing’ group, and a ‘pain physiology education’ group as the control group [ 49 ]. Two studies included educational sessions provided by a therapist plus activity monitoring via ActiGraph accelerometers [ 51 ] and diaries [ 48 ] at baseline and follow-up. In the first of these two studies, Nijs and colleagues [ 51 ] implemented a ‘self-management program’ which asked patients to estimate their current physical capabilities prior to commencing an activity and then complete 25–50% less than their perceived energy envelope. They[ 51 ] did not include a control group and had a sample size of only n = 7. Six years later, the same research group [ 48 ] conducted another pacing study which utilised relaxation as a comparator group (n = 12 and n = 14 in the pacing and relaxation groups, respectively). The pacing group underwent a pacing phase whereby participants again aimed to complete 25–50% less than their perceived energy envelope, followed by a gradual increase in exercise after the pacing phase (the total intervention spanned three weeks, and it is unclear how much was allocated to pacing, and how much to activity increase). Therefore, it could be argued that Kos et al. [ 48 ] really assessed pacing followed by a gradual exercise increase as outcome measures were assessed following the graded activity phase. Another pacing intervention delivered weekly educational sessions for six weeks and utilised a standardised rehabilitation programme using the ‘activity pacing framework’ [ 50 ] in a single-arm, no comparator group feasibility study. Finally, the PACE trial adopted an adaptive pacing therapy intervention consisting of occupational therapists helping patients to plan and pace activities utilising activity diaries to identify activities associated with fatigue and staying within their energy envelope [ 35 ]. This study incorporated standard medical care, cognitive behavioural therapy (CBT) and graded exercise therapy (GET) as comparator groups [ 35 ]. It is worth noting that the pacing group and the CBT group were both ‘encouraged’ to increase physical activity levels as long as participants did not exceed their energy envelope. Although not all five intervention studies explicitly mentioned the “Energy Envelope Theory”, which dictates that people with ME/CFS should not necessarily increase or decrease their activity levels, but moderate activity and practice energy conservation [ 62 ], all intervention studies used language analogous to this theory, such as participants staying within limits, within capacity, or similar.

The interventions included in this review were of varying durations, from a single 30-min education session [ 49 ], a 3-week (one session a week) educational programme [ 51 ], a 3-week (3 × 60–90 min sessions/week) educational programme [ 48 ], a 6-week rehabilitation programme [ 50 ], to a 24-week programme [ 35 ]. Intervention follow-up durations also varied across studies from immediately after [ 49 ], 1-week [ 51 ], 3-weeks [ 48 ], 3-months [ 50 ], and 1-year post-intervention [ 35 ].

Observational studies of pacing

Eight studies were observational and, therefore, included no intervention. Observational study sample sizes ranged from 16 in a cross-sectional interview study [ 25 ] to 1428 in a cross-sectional survey [ 52 ]. One study involved a retrospective analysis of participants’ own pacing strategies varying from self-guided pacing or pacing administered by a therapist compared with implementation of CBT and GET [ 52 ]. Five involved a cross-sectional analysis of participants own pacing strategies which varied from activity adjustment, planning and acceptance [ 50 , 55 ], and the Energy Envelope method [ 58 , 60 ]. Two studies were prospective observational studies investigating the Energy Envelope theory [ 53 , 54 ]. Four studies [ 56 , 57 , 59 , 61 ] included in this review involved sub-analysis of results of the PACE trial [ 35 ].

Outcome measures

Quantitative health outcomes.

ME/CFS severity and general health status were the most common outcome measures across studies (16/17) [ 35 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 , 61 , 63 ]. Studies utilised different instruments, including the Short-Form 36 (SF-36; 8/16) [ 35 , 51 , 53 , 54 , 56 , 57 , 58 , 60 ], SF-12 (2/16) [ 50 , 63 ], ME symptom and illness severity (2/16) [ 52 , 55 ], Patient health (PHQ-15; 1/16) [ 59 ], DePaul symptom questionnaire (DSQ; 1/16) [ 58 ], and the Patient health questionnaire-9 (1/16) [ 50 ]. Additionally, some studies used diagnostic criteria for ME/CFS as an outcome measure to determine recovery [ 57 , 59 , 61 ].

Pain was assessed by most included studies (11/17) [ 35 , 49 , 50 , 51 , 53 , 54 , 55 , 57 , 59 , 60 , 61 , 63 ]. Two studies [ 59 , 61 ] included the international CDC criteria for CFS which contain five painful symptoms central to a diagnosis of CFS: muscle pain and joint pain. Other methods of assessment included Brief Pain Inventory (1/11) [ 53 ], Chronic Pain Coping Inventory (CPCI; 1/11) [ 49 ], Pain Self Efficacy Questionnaire (PSEQ; 1/11) [ 50 ], Tampa Scale for Kinesiophobia–version CFS (1/11) [ 49 ], algometry (1/11) [ 49 ], Knowledge of Neurophysiology of Pain Test (1/12) [ 49 ], Pain Catastrophizing Scale (1/11) [ 49 ], Pain Anxiety Symptoms Scale short version (PASS-20; 1/11) [ 50 ], Pain Numerical Rating Scale (NRS; 1/11) [ 63 ].

Fatigue or post-exertional malaise was assessed by 11 of the 17 studies [ 35 , 48 , 50 , 51 , 53 , 54 , 56 , 57 , 60 , 61 , 63 ]. Again, measurement instruments were divergent between studies and included the Chalder Fatigue Questionnaire (CFQ; 4/11) [ 35 , 50 , 57 , 63 ], Fatigue Severity Scale (2/11) [ 53 , 60 ], the Chronic Fatigue Syndrome Medical Questionnaire (1/11) [ 60 ], and Checklist Individual Strength (CIS; 2/11) [ 48 , 51 ].

Anxiety and depression were also common outcome measures, utilised by four studies (4/17) [ 50 , 53 , 59 , 63 ]. These were also assessed using different instruments including Hospital Anxiety and Depression Scale (HADS; 2/4) [ 59 , 63 ], Generalised Anxiety Disorder Assessment (1/4 [ 50 ]), Beck Depression Inventory (BDI-II; 1/4) [ 53 ], Beck Anxiety Inventory (BAI; 1/4) [ 53 ], and Perceived Stress Scale (PSS; 1/4) [ 53 ].

Outcome measures also included sleep (2/17) [ 53 , 59 ], assessed by The Pittsburgh Sleep Quality Index (1/2) [ 53 ] and Jenkins sleep scale (1/2) [ 59 ]; and quality of life (2/17) [ 50 , 53 ] as assessed by the EuroQol five-dimensions, five-levels (EQ-5D-5L; 1/2) [ 50 ] and The Quality-of-Life Scale (1/2) [ 53 ]. Self-Efficacy was measured in four studies [ 50 , 53 , 59 , 60 ], assessed by the Brief Coping Orientation to Problems Experienced Scale (bCOPE; 1/4) [ 60 ] and the Chronic Disease Self-Efficacy measure (3/4) [ 50 , 53 , 59 ].

Quantitative evaluation of pacing

Some studies (4/17) [ 25 , 50 , 52 , 63 ] included assessments of the participants’ experiences of pacing, using the Activity Pacing Questionnaire (APQ-28; 1/4 [ 50 ], APQ-38 (2/4) [ 25 , 63 ]), a re-analysis of the 228 question survey regarding treatment (1/4) [ 52 ] originally produced by the ME Association [ 55 ], and qualitative semi-structured telephone interviews regarding appropriateness of courses in relation to individual patient needs (1/4) [ 25 ]. The APQ-28 and -38 have been previously validated, but the 228-question survey has not. When outcome measures included physical activity levels (4/17), the Canadian Occupational Performance Measure (COPM) was used in two studies [ 48 , 51 ], and two studies used accelerometers to record physical activity [ 51 , 54 ]. Of these two studies, Nijs [ 51 ] examined accelerometery after a 3-week intervention based on the Energy Envelope Theory and Brown et al. [ 54 ] evaluated the Energy Envelope Theory of pacing over 12 months.

Other outcomes

Two [ 53 , 59 ] of the 17 studies included structured clinical interviews for the Diagnostic and Statistical Manual of Mental Disorders, 4th edition (DSM-IV) to assess psychiatric comorbidity and psychiatric exclusions. One study included a disability benefits questionnaire [ 55 ], and one study included employment and education questionnaire [ 55 ]. Additionally, satisfaction of primary care was also used as an outcome measure (2/17) [ 25 , 55 ] assessed using the Chronic Pain Coping Inventory (CPCI).

Efficacy of pacing interventions

The majority of studies (12/17) [ 25 , 48 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 58 , 60 , 63 ] highlighted improvements in at least one outcome following pacing (Fig.  2 ). When the effect of pacing was assessed by ME symptomology and general health outcomes, studies reported pacing to be beneficial [ 25 , 50 , 51 , 53 , 54 , 55 , 56 , 58 ]. It is worth noting however that pacing reportedly worsened ME symptoms in 14% of survey respondents, whilst improving symptoms in 44% of respondents [ 52 ]. Most studies using fatigue as an outcome measure reported pacing to be efficacious (7/10) [ 50 , 51 , 53 , 54 , 56 , 60 , 63 ]. However, one study reported no change in fatigue with a pacing intervention (1/10) [ 35 ], and 2/10 studies [ 53 , 63 ] reported a worsening of fatigue with pacing. Physical function was used to determine the efficacy of pacing in 11 studies [ 35 , 48 , 50 , 51 , 53 , 54 , 56 , 58 , 59 , 60 , 63 ]. Of these, the majority found pacing improved physical functioning (8/10) [ 48 , 50 , 51 , 53 , 54 , 56 , 58 , 60 ], with 1/10 [ 35 ] studies reporting no change in physical functioning, and 1/10 [ 59 ] reporting a worsening of physical functioning from pre- to post-pacing. Of the seven studies [ 35 , 49 , 50 , 51 , 53 , 54 , 60 ] which used pain to assess pacing efficacy, 4/7 [ 50 , 51 , 53 , 60 ] reported improvements in pain and 3/7 [ 35 , 51 , 53 ] reported no change in pain scores with pacing. All studies reporting quality of life (1/1) [ 53 ], self-efficacy (3/3) [ 50 , 53 , 59 ], sleep (2/2) [ 53 , 59 ], and depression and anxiety (4/4) [ 50 , 53 , 59 , 63 ], found pacing to be efficacious for ME/CFS participants.

Bubble plot displaying number of studies reporting each domain (x-axis) and the percentage of studies reporting improvement with pacing (y-axis), including a coloured scale of improvement from 0–100%. PEM = post-exertional malaise, 6MWT = 6-min walk time, CFS = chronic fatigue syndrome, DSQ = DePaul Symptom Questionnaire, PA = Physical Activity, HRQOL = Health-related quality of life, COPM = The Canadian Occupational Performance Measure

Participant characteristics

The majority of studies (10/17) [ 25 , 50 , 52 , 53 , 54 , 58 , 59 , 60 , 61 , 63 ] did not report age of the participants. For those which did report age, this ranged from 32 ± 14 to 43 ± 13 years. Where studies reported sex (11/17) [ 35 , 48 , 49 , 50 , 51 , 54 , 55 , 56 , 57 , 58 , 60 ], this was predominantly female, ranging from 75 to 100% female. Only six studies [ 35 , 54 , 56 , 57 , 58 , 60 ] reported ethnicity, with cohorts predominantly Caucasian (94–98%). Time since diagnosis was mostly unreported (12/17) [ 25 , 48 , 49 , 50 , 52 , 53 , 54 , 58 , 59 , 60 , 61 , 63 ] but ranged from 32 to 96 months, with a cross-sectional survey reporting 2% of the participants were diagnosed 1–2 years previously; 6% 3–4 years since diagnosis; 13% 3–4 years since diagnosis; 12% 5–6 years since diagnosis; 20% 7–10 years since diagnosis; 29% 11–21 years since diagnosis; 13% 21–30 years since diagnosis; and 5% > 30 years since diagnosis. Of the studies which reported comorbidities of the participants (6/17) [ 25 , 35 , 50 , 56 , 57 , 63 ], the comorbidities were chronic pain, depressive disorder, psychiatric disorder.

Study location

Of the 17 studies, 14 were from Europe [ 25 , 35 , 48 , 49 , 50 , 51 , 52 , 55 , 56 , 57 , 58 , 59 , 61 , 63 ], and three from North America [ 53 , 54 , 60 ]. Of the 14 studies[ 25 , 35 , 48 , 49 , 50 , 51 , 52 , 55 , 56 , 57 , 58 , 59 , 61 , 63 ] from Europe, ten [ 25 , 35 , 50 , 52 , 55 , 56 , 57 , 58 , 59 , 61 , 63 ] were conducted in the United Kingdom, three in Belgium [ 48 , 49 , 51 ], and one was a multicentred study between the United Kingdom and Norway [ 58 ].

Recruitment strategy

Of the 17 studies, three [ 53 , 54 , 60 ] used announcements in a newspaper and physician referrals to recruit participants, two [ 50 , 63 ] recruited patients referred by a consultant from a National Health Service (NHS) Trust following a pain diagnosis, two [ 52 , 55 ] concerned online platforms on the web, two [ 59 , 61 ] recruited from secondary care clinics, and two used the PACE trial databases [ 56 , 57 ]. Moreover, one study recruited from the hospital [ 58 ], one from physiotherapist referrals [ 25 ], two from specialist clinic centres [ 35 , 64 ], one from waiting list of rehabilitation centre [ 48 ], and one from medical files [ 49 ].

Study settings

Ten studies were carried out in hospital and clinic setting [ 25 , 35 , 48 , 49 , 50 , 51 , 58 , 59 , 61 , 63 ]. Two studies were performed on online platforms [ 52 , 55 ]. Three studies did not report study setting [ 53 , 54 , 60 ]. Two studies generated output from PACE trial databases [ 56 , 57 ]

Adherence and feasibility

All five intervention studies reported adherence rates (which they defined as number of sessions attended), which ranged from 4–44% (4% [ 49 ], 8% [ 35 ], 25% [ 48 ], 29% [ 51 ], and 44% [ 50 ]). One study reported the median number of rehabilitation programme sessions attended was five out of six possible sessions, with 58.9% [ 50 ] participants attending ≥ 5 sessions; 83.2% participants attending at least one educational session on activity pacing and 56.1% attending both activity pacing sessions.

This scoping review summarises the existing literature, with a view to aid physicians and healthcare practitioners better summarise evidence for pacing in ME/CFS and use this knowledge for other post-viral fatiguing conditions. Overall, studies generally reported pacing to be beneficial for people with ME/CFS. The exception to this trend is the controversial PACE trial [ 36 , 37 , 38 , 39 ], which we will expand on in subsequent sections. We believe information generated within this review can facilitate discussion of research opportunities and issues that need to be addressed in future studies concerning pacing, particularly given the immediate public health issue of the long COVID pandemic. As mentioned, we found some preliminary evidence for improved symptoms following pacing interventions or strategies. However, we wish to caution the reader that the current evidence base is extremely limited and hampered by several limitations which preclude clear conclusions on the efficacy of pacing. Firstly, studies were of poor to fair methodological quality (indicated by the PEDro scores), often with small sample sizes, and therefore unknown power to detect change. Moreover, very few studies implemented pacing, with most studies merely consulting on people’s views on pacing. This may of course lead to multiple biases such as reporting, recruitment, survivorship, confirmation, availability heuristic, to name but a few. Thus, there is a pressing need for more high-quality intervention studies. Secondly, the reporting of pacing strategies used was inconsistent and lacked detail, making it difficult to describe current approaches, or implement them in future research or symptom management strategies. Furthermore, outcome evaluations varied greatly between studies. This prevents any appropriate synthesis of research findings.

The lack of evidence concerning pacing is concerning given pacing is the only NICE recommended management strategy for ME/CFS following the 2021 update [ 34 ]. Given the analogous nature of long COVID with ME/CFS, patients and practitioners will be looking to the ME/CFS literature for guidance for symptom management. There is an urgent need for high quality studies (such as RCTs) investigating the effectiveness of pacing and better reporting of pacing intervention strategies so that clear recommendations can be made to patients. If this does not happen soon, there will be serious healthcare and economic implications for years to come [ 65 , 66 ].

Efficacy of pacing

Most studies (12/17) highlighted improvements in at least one outcome measure following pacing. Pacing was self-reported to be the most efficacious, safe, acceptable, and preferred form of activity management for people with ME/CFS [ 55 ]. Pacing was reported to improve symptoms and improve general health outcomes [ 25 , 50 , 52 , 58 , 63 ], fatigue and PEM [ 48 , 50 , 51 , 53 , 54 , 55 , 56 , 60 , 63 ], physical functioning [ 48 , 50 , 51 , 53 , 56 , 58 , 60 , 63 ], pain [ 25 , 50 , 55 , 63 ], quality of life [ 50 ], self-efficacy [ 50 , 53 ], sleep [ 53 , 55 ], and depression and anxiety [ 50 , 53 , 63 ]. These positive findings provide hope for those with ME/CFS, and other chronic fatiguing conditions such as long COVID, to improve quality of life through symptom management.

Conversely, some studies reported no effects of pacing on ME/CFS symptoms [ 52 ], fatigue, physical functioning [ 35 ], or pain scores [ 49 , 61 ]. Some studies even found pacing to have detrimental effects in those with ME/CFS, including a worsening of symptoms in 14% of survey participants recalling previous pacing experiences [ 52 ]. Furthermore, a worsening of fatigue [ 35 , 59 ], and physical functioning from pre- to post-pacing [ 35 , 57 , 59 , 61 ] was reported by the PACE trial and sub-analysis of the PACE trial [ 56 , 57 , 61 ]. The PACE trial [ 35 ], a large RCT (n = 639) comparing pacing with CBT and GET, reported GET and CBT were more effective for reducing ME/CFS-related fatigue and improving physical functioning than pacing. However, the methodology and conclusions from the PACE trial have been heavily criticised, mainly due to the authors lowering the thresholds they used to determine improvement [ 36 , 37 , 38 , 67 ]. With this in mind, Sharpe et al. [ 56 ] surveyed 75% of the participants from the PACE trial 1-year post-intervention and reported pacing improved fatigue and physical functioning, with effects similar to CBT and GET.

Lessons for pacing implementation

All pacing intervention studies (5/5) implemented educational or coaching sessions. These educational components were poorly reported in terms of the specific content and how and where they had been developed, with unclear pedagogical approaches. Consequently, even where interventions reported reduction in PEM or improved symptoms, it would be impossible to transfer that research into practice, future studies, or clinical guidance, given the ambiguity of reporting. Sessions typically contained themes of pacing such as activity adjustment (decrease, break-up, and reschedule activities based on energy levels), activity consistency (maintaining a consistently low level of activity to prevent PEM), activity planning (planning activities and rest around available energy levels), and activity progression (slowly progressing activity once maintaining a steady baseline) [ 35 , 48 , 49 , 50 , 51 ]. We feel it is pertinent to note here that although activity progression has been incorporated as a pacing strategy in these included studies, some view activity progression as a form of GET. The NICE definition of GET is “first establishing an individual's baseline of achievable exercise or physical activity, then making fixed incremental increases in the time spent being physically active” [ 34 ]. Thus, this form of pacing can also be considered a type of ‘long-term GET’ in which physical activity progression is performed over weeks or months with fixed incremental increases in time spent being physically.

Intervention studies attempted to create behaviour change, through educational programmes to modify physical activity, and plan behaviours. However, none of these studies detailed integrating any evidence-based theories of behaviour change [ 68 ] or reported using any frameworks to support behaviour change objectives. This is unfortunate since there is good evidence that theory-driven behaviour change interventions result in greater intervention effects [ 69 ]. Indeed, there is a large body of work regarding methods of behaviour change covering public health messaging, education, and intervention design, which has largely been ignored by the pacing literature. Interventions relied on subjective pacing (5/5 studies), with strategies including keeping an activity diary (3/5 studies) to identify links between activity and fatigue [ 35 , 48 , 50 ]. Given the high prevalence of ‘brain fog’ within ME/CFS [ 70 , 71 , 72 , 73 ], recall may be extremely difficult and there is significant potential for under-reporting. Other strategies included simply asking participants to estimate energy levels available for daily activities (2/5 studies [ 48 , 51 ]). Again, this is subjective and relies on participants’ ability to recall previous consequences of the activity. Other methods of activity tracking and measuring energy availability, such as wearable technology [ 74 , 75 , 76 , 77 , 78 ] could provide a more objective measure of adherence and pacing strategy fidelity in future studies. Despite technology such as accelerometers being widely accessible since well-before the earliest interventional study included in this review (which was published in 2009), none of the interventional studies utilised objective activity tracking to track pacing and provide feedback to participants. One study considered accelerometery alongside an activity diary [ 51 ]. However, accelerometery was considered the outcome variable, to assess change in activity levels from pre- to post-intervention and was not part of the intervention itself (which was one pacing coaching sessions per week for 3 weeks). Moreover, most research-grade accelerometers cannot be used as part of the intervention since they have no ability to provide continuous feedback and must be retrieved by the research team in order to access any data. Consequently, their use is mostly limited to outcome assessments only. As pacing comprises a limit to physical activity to prevent push-crash cycles, it is an astonishing observation from this scoping review that only two studies objectively measured physical activity to quantify changes to activity as a result of pacing [ 51 , 54 ]. If the aim of pacing is to reduce physical activity, or reduce variations in physical activity (i.e., push-crash cycles), only two studies have objectively quantified the effect pacing had on physical activity, so it is unclear whether pacing was successfully implemented in any of the other studies.

By exploring the pacing strategies previously used, in both intervention studies and more exploratory studies, we can identify and recommend approaches to improve symptoms of ME/CFS. These approaches can be categorised as follows: activity planning, activity consistency, activity progression, activity adjustment and staying within the Energy Envelope [ 50 , 53 , 60 , 63 ]. Activity planning was identified as a particularly effective therapeutic strategy, resulting in improvement of mean scores of all symptoms included in the APQ-28, reducing current pain, improvement of physical fatigue, mental fatigue, self-efficacy, quality of life, and mental and physical functioning [ 50 ]. Activity planning aligns with the self-regulatory behaviour change technique ‘Action Planning’ [ 79 ] which is commonly used to increase physical activity behaviour. In the case of ME/CFS, activity planning is successfully used to minimise rather than increase physical activity bouts to prevent expending too much energy and avoid PEM. Activity consistency, meaning undertaking similar amounts of activity each day, was also associated with reduced levels of depression, exercise avoidance, and higher levels of physical function [ 63 ]. Activity progression was associated with higher levels of current pain. Activity adjustment associated with depression and avoidance, and lower levels of physical function [ 63 ]. Staying within the Energy Envelope was reported to reduce PEM severity [ 53 , 60 ], improve physical functioning [ 53 , 60 ] and ME/CFS symptom scores [ 53 ], and more hours engaged in activity than individuals with lower available energy [ 53 ]. These results suggest that effective pacing strategies would include activity planning, consistency, and energy management techniques while avoiding progression. This data is, of course, limited by the small number of mostly low-quality studies and should be interpreted with some caution. Nevertheless, these are considerations that repeatedly appear in the literature and, as such, warrant deeper investigation. In addition, and as outlined earlier, most studies are relatively old, and we urgently need better insight into how modern technologies, particularly longitudinal activity tracking and contemporaneous heart-rate feedback, might improve (or otherwise) adaptive pacing. Such longitudinal tracking would also enable activities and other behaviours (sleep, diet, stress) to be linked to bouts of PEM. Linking would enable a deeper insight into potential PEM triggers and mitigations that might be possible.

The PACE trial

We feel it would be remiss of us to not specifically address the PACE trial within this manuscript, as five of the 17 included studies resulted from the PACE trial [ 35 , 56 , 57 , 59 , 61 ]. There has been considerable discussion around the PACE trial, which has been particularly divisive and controversial [ 37 , 38 , 39 , 59 , 67 , 80 , 81 ]. In the PACE trial, GET and CBT were deemed superior to pacing by the authors. Despite its size and funding, the PACE trial has received several published criticisms and rebuttals. Notably, NICE's most recent ME/CFS guideline update removed GET and CBT as suggested treatment options, which hitherto had been underpinned by the PACE findings. While we will not restate the criticisms and rebuttals here, what is not in doubt, is that the PACE trial has dominated discussions of pacing, representing almost a third of all the studies in this review. However, the trial results were published over a decade ago, with the study protocol devised almost two decades ago [ 82 ]. The intervening time has seen a revolution in the development of mobile and wearable technology and an ability to remotely track activity and provide real-time feedback in a way which was not available at that time. Furthermore, there has been no substantive research since the PACE trial that has attempted such work. Indeed, possibly driven by the reported lack of effect of pacing in the PACE trial, this review has demonstrated the dearth of progress and innovation in pacing research since its publication. Therefore, regardless of its findings or criticisms, the pacing implementation in the PACE trial is dated, and there is an urgent need for more technologically informed approaches to pacing research.

Limitations of the current evidence

The first limitation to the literature included in this scoping review is that not all studies followed the minimum data set (MDS) of patient-reported outcome measures (PROMs) agreed upon by the British Association of CFS/ME Professionals (BACME) (fatigue, sleep quality, self-efficacy, pain/discomfort, anxiety/depression, mobility, activities of daily living, self-care, and illness severity) [ 83 , 84 ]. All but one study included in this review measured illness severity, most studies included fatigue and pain/discomfort, and some studies included assessments of anxiety/depression. There was a lack of quantitative assessment of sleep quality, self-efficacy, mobility, activities of daily living, and self-care. Therefore, studies did not consistently capture the diverse nature of the symptoms experienced, with crucial domains missing from the analyses. The MDS of PROMs were established in 2012 [ 83 , 84 ] and therefore, for studies published out prior to 2012, these are not applicable [ 35 , 49 , 51 , 53 , 54 ]. However, for the 12 studies carried out after this time, the MDS should have been considered elucidate the effects of pacing on ME/CFS. Importantly, despite PEM being a central characteristic of ME/CFS, only two studies included PEM as an outcome measure [ 55 , 60 ]. This may be because of the difficulty of accurately measuring fluctuating symptoms, as PEM occurs multiple times over a period of months, and therefore pre- to post- studies and cross-sectional designs cannot adequately capture PEM incidence. Therefore, it is likely studies opted for measuring general fatigue instead. More appropriate longitudinal study designs are required to track PEM over time to capture a more representative picture of PEM patterns. Secondly, reporting of participant characteristics was inadequate, but in the studies that did describe participants, characteristics were congruent with the epidemiological literature and reporting of ME/CFS populations (i.e., 60–65% female) [ 85 ]. Therefore, in this respect, studies included herein were representative samples. However, the lack of reporting of participant characteristics limits inferences we can draw concerning any population-related effects (i.e. whether older, or male, or European, or people referred by a national health service would be more or less likely to respond positively to pacing). Thirdly, comparison groups (where included) were not ideal, with CBT or GET sometimes used as comparators to pacing [ 35 ], and often no true control group included. Penultimately, there is a distinct lack of high-quality RCTs (as mentioned throughout this manuscript). Finally, in reference to the previous section, inferences from the literature are dated and do not reflect the technological capabilities of 2023.

Recommendations for advancement of the investigative area

It is clear from the studies included in this scoping review for the last decade or more, progress and innovation in pacing research have been limited. This is unfortunate for several reasons. People with ME/CFS or long COVID are, of course, invested in their recovery. From our patient and public involvement (PPI) group engagement, it is clear many are ahead of the research and are using wearable technology to track steps, heart rate, and, in some cases, heart rate variability to improve their own pacing practice. While the lack of progress in the research means this is an understandable response by patients, it is also problematic. Without underpinning research, patients may make decisions based on an individual report of trial-and-error approaches given the lack of evidence-based guidance.

A more technologically-informed pacing approach could be implemented by integrating wearable trackers [ 77 , 78 , 86 , 87 ] to provide participants with live updates on their activity and could be integrated with research-informed messaging aimed at supporting behaviour change, as has been trialled in other research areas [ 88 , 89 , 90 , 91 ]. However, more work is needed to evaluate how to incorporate wearable activity trackers and which metrics are most helpful.

A more technologically-informed approach could also be beneficial for longitudinal symptom tracking, particularly useful given the highly variable symptom loads of ME/CFS and episodic nature of PEM. This would overcome reliance on assessments at a single point in time (as the studies within this review conducted). Similarly, mobile health (mHealth) approaches also allow questionnaires to be digitised to make it easier for participants to complete if they find holding a pen or reading small font problematic [ 92 ]. Reminders and notifications can also be helpful for patients completing tasks [ 77 , 93 , 94 , 95 ]. This approach has the added advantage of allowing contemporaneous data collection rather than relying on pre- to post-intervention designs limited by recall bias. Future work must try to leverage these approaches, as unless we collect large data sets on symptoms and behaviours (i.e. activity, diet, sleep, and pharmacology) in people with conditions like ME/CFS we will not be able to leverage emerging technologies such as AI and machine learning to improve the support and care for people with these debilitating conditions. The key areas for research outline in the NICE guidelines (2021 update) speaks to this, with specific mention of improved self-monitoring strategies, sleep strategies, and dietary strategies, all of which can be measured using mHealth approaches, in a scalable and labour-inexpensive way.

The potential for existing pacing research to address the long COVID pandemic

There is now an urgent public health need to address long COVID, with over 200 million sufferers worldwide [ 30 ]. Given the analogous symptomology between ME/CFS and long COVID, and the lack of promising treatment and management strategies in ME/CFS, pacing remains the only strategy for managing long COVID symptoms. This is concerning as the quality of evidence to support pacing is lacking. Given long COVID has reached pandemic proportions, scalable solutions will be required. In this context, we propose that technology should be harnessed to a) deliver, but also b) evaluate, pacing. We recently reported on a just-in-time adaptive intervention to increase physical activity during the pandemic [ 78 ]. However, this method could be adapted to decrease or maintain physical activity levels (i.e., pacing) in long COVID. This method has the advantage of scalability and remote data collection, reducing resource commitments and participant burden, essential for addressing a condition with so many sufferers.

This review highlights the need for more studies concerning pacing in chronic fatiguing conditions. Future studies would benefit from examining pacing’s effect on symptomology and PEM with objectively quantified pacing, over a longer duration of examination, using the MDS. It is essential this is conducted as an RCT, given that in the case of long COVID, participants may improve their health over time, and it is necessary to determine whether pacing exerts an additional effect over time elapsing. Future studies would benefit from digitising pacing to support individuals with varying symptom severity and personalise support. This would improve accessibility and reduce selection bias, in addition to improving scalability of interventions. Finally, clinicians and practitioners should be cognisant of the strength of evidence reported in this review and should exert caution when promoting pacing in their patients, given the varying methods utilised herein.

Availability of data and materials

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

Abbreviations

Activity Pacing Questionnaire

Beck Anxiety Inventory

Beck Depression Inventory

Brief Coping Orientation to Problems Experienced Scale

Canadian Occupational Performance Measure

Centers for disease control and prevention

Chalder Fatigue Questionnaire

Checklist Individual Strength

Chronic Pain Coping Inventory

Cognitive behavioural therapy

Cochrane Central Register of Controlled Trials

DePaul symptom questionnaire

EuroQol five-dimensions, five-levels questionnaire

Graded exercise therapy

Hospital Anxiety and Depression Scale

Myalgic encephalomyelitis/chronic fatigue syndrome

Pain Self Efficacy Questionnaire

Pain Anxiety Symptoms Scale short version

Pain Numerical Rating Scale

Patient health questionnaire

Patient reported outcome measures

Physiotherapy Evidence Database

Perceived Stress Scale

Post exertional malaise

Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for scoping reviews

Randomised control trial

McMurray JC, May JW, Cunningham MW, Jones OY. Multisystem Inflammatory Syndrome in Children (MIS-C), a post-viral myocarditis and systemic vasculitis-a critical review of its pathogenesis and treatment. Front Pediatr. 2020;8: 626182.

Article   PubMed   PubMed Central   Google Scholar  

Perrin R, Riste L, Hann M, Walther A, Mukherjee A, Heald A. Into the looking glass: post-viral syndrome post COVID-19. Med Hypotheses. 2020;144: 110055.

Article   PubMed   PubMed Central   CAS   Google Scholar  

Hayes LD, Ingram J, Sculthorpe NF. More than 100 persistent symptoms of SARS-CoV-2 (Long COVID): A scoping review. Front Med. 2021. https://doi.org/10.3389/fmed.2021.750378 .

Article   Google Scholar  

McLaughlin M, Cerexhe C, Macdonald E, Ingram J, Sanal-Hayes NEM, Hayes LD, et al. A Cross-sectional study of symptom prevalence, frequency, severity, and impact of long-COVID in Scotland: part I. Am J Med. 2023. https://doi.org/10.1016/j.amjmed.2023.07.009 .

Article   PubMed   Google Scholar  

McLaughlin M, Cerexhe C, Macdonald E, Ingram J, Sanal-Hayes NEM, Hayes LD, et al. A cross-sectional study of symptom prevalence, frequency, severity, and impact of long-COVID in Scotland: part II. Am J Med. 2023. https://doi.org/10.1016/j.amjmed.2023.07.009 .

Hayes LD, Sanal-Hayes NEM, Mclaughlin M, Berry ECJ, Sculthorpe NF. People with long covid and ME/CFS exhibit similarly impaired balance and physical capacity: a case-case-control study. Am J Med. 2023;S0002–9343(23):00465–75.

Google Scholar  

Jenkins R. Post-viral fatigue syndrome. Epidemiology: lessons from the past. Br Med Bull. 1991;47:952–65.

Article   PubMed   CAS   Google Scholar  

Sandler CX, Wyller VBB, Moss-Morris R, Buchwald D, Crawley E, Hautvast J, et al. Long COVID and post-infective fatigue syndrome: a review. Open Forum Infect Dis. 2021;8:440.

Carod-Artal FJ. Post-COVID-19 syndrome: epidemiology, diagnostic criteria and pathogenic mechanisms involved. Rev Neurol. 2021;72:384–96.

PubMed   CAS   Google Scholar  

Ballering AV, van Zon SKR, Olde Hartman TC, Rosmalen JGM. Lifelines corona research initiative. Persistence of somatic symptoms after COVID-19 in the Netherlands: an observational cohort study. Lancet. 2022;400:452–61.

Wong TL, Weitzer DJ. Long COVID and Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS)-a systemic review and comparison of clinical presentation and symptomatology. Medicina (Kaunas). 2021;57:418.

Sukocheva OA, Maksoud R, Beeraka NM, Madhunapantula SV, Sinelnikov M, Nikolenko VN, et al. Analysis of post COVID-19 condition and its overlap with myalgic encephalomyelitis/chronic fatigue syndrome. J Adv Res. 2021. https://doi.org/10.1016/j.jare.2021.11.013 .

Bonilla H, Quach TC, Tiwari A, Bonilla AE, Miglis M, Yang P, et al. Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) is common in post-acute sequelae of SARS-CoV-2 infection (PASC): results from a post-COVID-19 multidisciplinary clinic. medrxiv. 2022. https://doi.org/10.1101/2022.08.03.22278363v1 .

Twomey R, DeMars J, Franklin K, Culos-Reed SN, Weatherald J, Wrightson JG. Chronic fatigue and postexertional malaise in people living with long COVID: an observational study. Phys Ther. 2022;102:005.

Barhorst EE, Boruch AE, Cook DB, Lindheimer JB. Pain-related post-exertional malaise in myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) and fibromyalgia: a systematic review and three-level meta-analysis. Pain Med. 2022;23:1144–57.

Goudsmit EM. The psychological aspects and management of chronic fatigue syndrome [Internet] [Thesis]. Brunel University, School of Social Sciences; 1996 [cited 2022 Jan 20]. https://scholar.google.co.uk/scholar_url?url=https://bura.brunel.ac.uk/bitstream/2438/4283/1/FulltextThesis.pdf&hl=en&sa=X&ei=kNYjZdeuA4-8ywTAmKmADQ&scisig=AFWwaeZvdxcuHmzGL08L3jp-QwNn&oi=scholarr . Accessed 2 Aug 2022

Stussman B, Williams A, Snow J, Gavin A, Scott R, Nath A, et al. Characterization of post-exertional malaise in patients with myalgic encephalomyelitis/chronic fatigue syndrome. Front Neurol. 2020;11:1025.

Holtzman CS, Bhatia KP, Cotler J, La J. Assessment of Post-Exertional Malaise (PEM) in Patients with Myalgic Encephalomyelitis (ME) and Chronic Fatigue Syndrome (CFS): a patient-driven survey. Diagnostics. 2019. https://doi.org/10.3390/diagnostics9010026 .

Fukuda K, Straus SE, Hickie I, Sharpe MC, Dobbins JG, Komaroff A. The chronic fatigue syndrome: a comprehensive approach to its definition and study. International Chronic Fatigue Syndrome Study Group. Ann Intern Med. 1994;121:953–9.

Carruthers BM, van de Sande MI, De Meirleir KL, Klimas NG, Broderick G, Mitchell T, et al. Myalgic encephalomyelitis: international consensus criteria. J Intern Med. 2011;270:327–38.

Carruthers JD, Lowe NJ, Menter MA, Gibson J, Eadie N, Botox Glabellar Lines II Study Group. Double-blind, placebo-controlled study of the safety and efficacy of botulinum toxin type A for patients with glabellar lines. Plast Reconstr Surg. 2003;112:1089–98.

Jason LA, Jordan K, Miike T, Bell DS, Lapp C, Torres-Harding S, et al. A pediatric case definition for myalgic encephalomyelitis and chronic fatigue syndrome. J Chronic Fatigue Syndrome. 2006;13:1–44.

Goudsmit EM, Nijs J, Jason LA, Wallman KE. Pacing as a strategy to improve energy management in myalgic encephalomyelitis/chronic fatigue syndrome: a consensus document. Disabil Rehabil. 2012;34:1140–7.

Antcliff D, Keenan A-M, Keeley P, Woby S, McGowan L. Engaging stakeholders to refine an activity pacing framework for chronic pain/fatigue: a nominal group technique. Musculoskeletal Care. 2019;17:354–62.

Antcliff D, Keeley P, Campbell M, Woby S, McGowan L. Exploring patients’ opinions of activity pacing and a new activity pacing questionnaire for chronic pain and/or fatigue: a qualitative study. Physiotherapy. 2016;102:300–7.

Yoshiuchi K, Cook DB, Ohashi K, Kumano H, Kuboki T, Yamamoto Y, et al. A real-time assessment of the effect of exercise in chronic fatigue syndrome. Physiol Behav. 2007;92:963–8.

Davenport TE, Stevens SR, Baroni K, Van Ness M, Snell CR. Diagnostic accuracy of symptoms characterising chronic fatigue syndrome. Disabil Rehabil. 2011;33:1768–75.

Nurek M, Rayner C, Freyer A, Taylor S, Järte L, MacDermott N, Delaney BC, Panellists D, et al. Recommendations for the recognition, diagnosis, and management of long COVID: a Delphi study. Br J Gen Pract. 2021. https://doi.org/10.3399/BJGP.2021.0265 .

Herrera JE, Niehaus WN, Whiteson J, Azola A, Baratta JM, Fleming TK, Kim SY, Naqvi H, Sampsel S, Silver JK, Gutierrez MV, Maley J, Herman E, Abramoff Benjamin, et al. Multidisciplinary collaborative consensus guidance statement on the assessment and treatment of fatigue in postacute sequelae of SARS-CoV-2 infection (PASC) patients. PM & R. 2021. https://doi.org/10.1002/pmrj.12684 .

Chen C, Haupert SR, Zimmermann L, Shi X, Fritsche LG, Mukherjee B. Global prevalence of post COVID-19 condition or long COVID: a meta-analysis and systematic review. J Infect Dis. 2022. https://doi.org/10.1093/infdis/jiac136 .

Office for National Statistics. Prevalence of ongoing symptoms following coronavirus (COVID-19) infection in the UK [Internet]. https://www.ons.gov.uk/peoplepopulationandcommunity/healthandsocialcare/conditionsanddiseases/bulletins/prevalenceofongoingsymptomsfollowingcoronaviruscovid19infectionintheuk/7july2022 . Accessed 2 Aug 2022

Office for National Statistics. Prevalence of ongoing symptoms following coronavirus (COVID-19) infection in the UK [Internet]. [cited 2022 Apr 1]. https://www.ons.gov.uk/peoplepopulationandcommunity/healthandsocialcare/conditionsanddiseases/bulletins/prevalenceofongoingsymptomsfollowingcoronaviruscovid19infectionintheuk/3march2022

Baker R, Shaw EJ. Diagnosis and management of chronic fatigue syndrome or myalgic encephalomyelitis (or encephalopathy): summary of NICE guidance. BMJ. 2007;335:446–8.

NICE. Overview | Myalgic encephalomyelitis (or encephalopathy)/chronic fatigue syndrome: diagnosis and management | Guidance | NICE [Internet]. NICE; [cited 2022 Aug 22]. https://www.nice.org.uk/guidance/ng206 . Accessed 2 Aug 2022

White P, Goldsmith K, Johnson A, Potts L, Walwyn R, DeCesare J, et al. Comparison of adaptive pacing therapy, cognitive behaviour therapy, graded exercise therapy, and specialist medical care for chronic fatigue syndrome (PACE): a randomised trial. The Lancet. 2011;377:823–36.

Article   CAS   Google Scholar  

Vink M. PACE trial authors continue to ignore their own null effect. J Health Psychol. 2017;22:1134–40.

Petrie K, Weinman J. The PACE trial: it’s time to broaden perceptions and move on. J Health Psychol. 2017;22:1198–200.

Stouten B. PACE-GATE: an alternative view on a study with a poor trial protocol. J Health Psychol. 2017;22:1192–7.

Agardy S. Chronic fatigue syndrome patients have no reason to accept the PACE trial results: response to Keith J Petrie and John Weinman. J Health Psychol. 2017;22:1206–8.

Kim D-Y, Lee J-S, Park S-Y, Kim S-J, Son C-G. Systematic review of randomized controlled trials for chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME). J Transl Med. 2020;18:7.

Twisk FNM, Maes M. A review on cognitive behavorial therapy (CBT) and graded exercise therapy (GET) in myalgic encephalomyelitis (ME) / chronic fatigue syndrome (CFS): CBT/GET is not only ineffective and not evidence-based, but also potentially harmful for many patients with ME/CFS. Neuro Endocrinol Lett. 2009;30:284–99.

PubMed   Google Scholar  

Mays N, Roberts E, Popay J. Synthesising research evidence. In: Fulop N, Allen P, Clarke A, Black N, editors. Studying the organisation and delivery of health services: research methods. London: Routledge; 2001. p. 188–220.

Munn Z, Peters MDJ, Stern C, Tufanaru C, McArthur A, Aromataris E. Systematic review or scoping review? Guidance for authors when choosing between a systematic or scoping review approach. BMC Med Res Methodol. 2018;18:143.

Arksey H, O’Malley L. Scoping studies: towards a methodological framework. Int J Soc Res Methodol. 2005;8:19–32.

Tricco AC, Lillie E, Zarin W, O’Brien KK, Colquhoun H, Levac D, et al. PRISMA extension for scoping reviews (PRISMA-ScR): checklist and explanation. Ann Intern Med. 2018;169:467–73.

de Morton NA. The PEDro scale is a valid measure of the methodological quality of clinical trials: a demographic study. Aust J Physiother. 2009;55:129–33.

Maher CG, Sherrington C, Herbert RD, Moseley AM, Elkins M. Reliability of the PEDro scale for rating quality of randomized controlled trials. Phys Ther. 2003;83:713–21.

Kos D, van Eupen I, Meirte J, Van Cauwenbergh D, Moorkens G, Meeus M, et al. Activity pacing self-management in chronic fatigue syndrome: a randomized controlled trial. Am J Occup Ther. 2015;69:6905290020.

Meeus M, Nijs J, Van Oosterwijck J, Van Alsenoy V, Truijen S. Pain physiology education improves pain beliefs in patients with chronic fatigue syndrome compared with pacing and self-management education: a double-blind randomized controlled trial. Arch Phys Med Rehabil. 2010;91:1153–9.

Antcliff D, Keenan A-M, Keeley P, Woby S, McGowan L. Testing a newly developed activity pacing framework for chronic pain/fatigue: a feasibility study. BMJ Open. 2021;11: e045398.

Nijs J, van Eupen I, Vandecauter J, Augustinus E, Bleyen G, Moorkens G, et al. Can pacing self-management alter physical behavior and symptom severity in chronic fatigue syndrome? A case series. J Rehabil Res Dev. 2009;46:985–96.

Geraghty K, Hann M, Kurtev S. Myalgic encephalomyelitis/chronic fatigue syndrome patients’ reports of symptom changes following cognitive behavioural therapy, graded exercise therapy and pacing treatments: Analysis of a primary survey compared with secondary surveys. J Health Psychol. 2019;24:1318–33.

Jason L, Muldowney K, Torres-Harding S. The energy envelope theory and myalgic encephalomyelitis/chronic fatigue syndrome. AAOHN J. 2008;56:189–95.

Brown M, Khorana N, Jason LA. The role of changes in activity as a function of perceived available and expended energy in non-pharmacological treatment outcomes for ME/CFS. J Clin Psychol. 2011;67:253.

Association ME. ME/CFS illness management survey results:‘“No decisions about me without me.” Part 1: Results and in-depth analysis of the 2012 ME association patient survey examining the acceptability, efficacy and safety of cognitive behavioural therapy, graded exercise therapy and pacing, as interventions used as management strategies for ME/CFS. 2015. https://www.meassociation.org.uk/wp-content/uploads/NO-DECISIONS-WITHOUT-ME-report.docx . Accessed 2 Feb 2022

Sharpe M, Goldsmith KA, Johnson AL, Chalder T, Walker J, White PD. Rehabilitative treatments for chronic fatigue syndrome: long-term follow-up from the PACE trial. The Lancet Psychiatry. 2015;2:1067–74.

White PD, Goldsmith K, Johnson AL, Chalder T, Sharpe M. Recovery from chronic fatigue syndrome after treatments given in the PACE trial. Psychol Med. 2013;43:2227–35.

O’connor K, Sunnquist M, Nicholson L, Jason LA, Newton JL, Strand EB. Energy envelope maintenance among patients with myalgic encephalomyelitis and chronic fatigue syndrome: Implications of limited energy reserves. Chronic Illn. 2019;15:51–60.

Dougall D, Johnson A, Goldsmith K, Sharpe M, Angus B, Chalder T, et al. Adverse events and deterioration reported by participants in the PACE trial of therapies for chronic fatigue syndrome. J Psychosom Res. 2014;77:20–6.

Brown AA, Evans MA, Jason LA. Examining the energy envelope and associated symptom patterns in chronic fatigue syndrome: does coping matter? Chronic Illn. 2013;9:302–11.

Bourke JH, Johnson AL, Sharpe M, Chalder T, White PD. Pain in chronic fatigue syndrome: response to rehabilitative treatments in the PACE trial. Psychol Med. 2014;44:1545–52.

Jason LA, Brown M, Brown A, Evans M, Flores S, Grant-Holler E, et al. Energy conservation/envelope theory interventions to help patients with myalgic encephalomyelitis/chronic fatigue syndrome. Fatigue. 2013;1:27–42.

Antcliff D, Campbell M, Woby S, Keeley P. Activity pacing is associated with better and worse symptoms for patients with long-term conditions. Clin J Pain. 2017;33:205–14.

Nijs T, Klein Y, Mousavi S, Ahsan A, Nowakowska S, Constable E, et al. The different faces of 4’-Pyrimidinyl-Functionalized 4,2’:6’,4"-Terpyridin es: metal-organic assemblies from solution and on Au(111) and Cu(111) surface platforms. J Am Chem Soc. 2018;140:2933–9.

Cutler DM, Summers LH. The COVID-19 pandemic and the $16 Trillion Virus. JAMA. 2020;324:1495–6.

Cutler DM. The costs of long COVID. JAMA Health Forum. 2022;3:e221809–e221809.

Geraghty K. ‘PACE-Gate’: when clinical trial evidence meets open data access. J Health Psychol. 2017;22:1106–12.

Davis R, Campbell R, Hildon Z, Hobbs L, Michie S. Theories of behaviour and behaviour change across the social and behavioural sciences: a scoping review. Health Psychol Rev. 2015;9:323–44.

Prestwich A, Sniehotta FF, Whittington C, Dombrowski SU, Rogers L, Michie S. Does theory influence the effectiveness of health behavior interventions? Meta-analysis Health Psychol. 2014;33:465–74.

Balinas C, Eaton-Fitch N, Maksoud R, Staines D, Marshall-Gradisnik S. Impact of life stressors on Myalgic encephalomyelitis/chronic fatigue syndrome symptoms: an Australian longitudinal study. Int J Environ Res Public Health. 2021;18:10614.

McGregor NR, Armstrong CW, Lewis DP, Gooley PR. Post-exertional malaise is associated with hypermetabolism, hypoacetylation and purine metabolism deregulation in ME/CFS cases. Diagnostics. 2019;9:70.

Nacul LC, Lacerda EM, Campion P, Pheby D, de Drachler M, Leite JC, et al. The functional status and well being of people with myalgic encephalomyelitis/chronic fatigue syndrome and their carers. BMC Public Health. 2011;11:402.

Deumer U-S, Varesi A, Floris V, Savioli G, Mantovani E, López-Carrasco P, et al. Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS): an overview. J Clin Med. 2021. https://doi.org/10.3390/jcm10204786 .

Düking P, Giessing L, Frenkel MO, Koehler K, Holmberg H-C, Sperlich B. Wrist-worn wearables for monitoring heart rate and energy expenditure while sitting or performing light-to-vigorous physical activity: validation study. JMIR Mhealth Uhealth. 2020;8: e16716.

Falter M, Budts W, Goetschalckx K, Cornelissen V, Buys R. Accuracy of apple watch measurements for heart rate and energy expenditure in patients with cardiovascular disease: cross-sectional study. JMIR Mhealth Uhealth. 2019;7: e11889.

Fuller D, Colwell E, Low J, Orychock K, Tobin MA, Simango B, et al. Reliability and validity of commercially available wearable devices for measuring steps, energy expenditure, and heart rate: systematic review. JMIR Mhealth Uhealth. 2020;8: e18694.

Mair JL, Hayes LD, Campbell AK, Sculthorpe N. Should we use activity tracker data from smartphones and wearables to understand population physical activity patterns? J Measur Phys Behav. 2022;1:1–5.

Mair JL, Hayes LD, Campbell AK, Buchan DS, Easton C, Sculthorpe N. A personalized smartphone-delivered just-in-time adaptive intervention (JitaBug) to increase physical activity in older adults: mixed methods feasibility study. JMIR Formative Res. 2022;6: e34662.

Michie S, Richardson M, Johnston M, Abraham C, Francis J, Hardeman W, et al. The behavior change technique taxonomy (v1) of 93 hierarchically clustered techniques: building an international consensus for the reporting of behavior change interventions. Ann Behav Med. 2013;46:81–95.

Feehan SM. The PACE trial in chronic fatigue syndrome. The Lancet. 2011;377:1831–2.

Giakoumakis J. The PACE trial in chronic fatigue syndrome. The Lancet. 2011;377:1831.

White PD, Sharpe MC, Chalder T, DeCesare JC, Walwyn R, PACE trial group. Protocol for the PACE trial: a randomised controlled trial of adaptive pacing, cognitive behaviour therapy, and graded exercise, as supplements to standardised specialist medical care versus standardised specialist medical care alone for patients with the chronic fatigue syndrome/myalgic encephalomyelitis or encephalopathy. BMC Neurol. 2007;7:6.

Reuben DB, Tinetti ME. Goal-oriented patient care–an alternative health outcomes paradigm. N Engl J Med. 2012;366:777–9.

Roberts D. Chronic fatigue syndrome and quality of life. PROM. 2018;9:253–62.

Valdez AR, Hancock EE, Adebayo S, Kiernicki DJ, Proskauer D, Attewell JR, et al. Estimating prevalence, demographics, and costs of ME/CFS using large scale medical claims data and machine learning. Front Pediatr. 2019. https://doi.org/10.3389/fped.2018.00412 .

Greiwe J, Nyenhuis SM. Wearable technology and how this can be implemented into clinical practice. Curr Allergy Asthma Rep. 2020;20:36.

Sun S, Folarin AA, Ranjan Y, Rashid Z, Conde P, Stewart C, et al. Using smartphones and wearable devices to monitor behavioral changes during COVID-19. J Med Internet Res. 2020;22: e19992.

Hardeman W, Houghton J, Lane K, Jones A, Naughton F. A systematic review of just-in-time adaptive interventions (JITAIs) to promote physical activity. Int J Behav Nutr Phys Act. 2019;16:31.

Perski O, Hébert ET, Naughton F, Hekler EB, Brown J, Businelle MS. Technology-mediated just-in-time adaptive interventions (JITAIs) to reduce harmful substance use: a systematic review. Addiction. 2022;117:1220–41.

AhmedS A, van Luenen S, Aslam S, van Bodegom D, Chavannes NH. A systematic review on the use of mHealth to increase physical activity in older people. Clinical eHealth. 2020;3:31–9.

Valenzuela T, Okubo Y, Woodbury A, Lord SR, Delbaere K. Adherence to technology-based exercise programs in older adults: a systematic review. J Geriatric Phys Ther. 2018;41:49–61.

Bowling A. Mode of questionnaire administration can have serious effects on data quality. J Public Health. 2005;27:281–91.

Burns SP, Terblanche M, Perea J, Lillard H, DeLaPena C, Grinage N, et al. mHealth intervention applications for adults living with the effects of stroke: a scoping review. Arch Rehabil Res Clin Transl. 2021;3: 100095.

Vandelanotte C, Müller AM, Short CE, Hingle M, Nathan N, Williams SL, et al. Past, present, and future of eHealth and mHealth research to improve physical activity and dietary behaviors. J Nutr Educ Behav. 2016;48:219-228.e1.

Ludwig K, Arthur R, Sculthorpe N, Fountain H, Buchan DS. Text messaging interventions for improvement in physical activity and sedentary behavior in youth: systematic review. JMIR Mhealth Uhealth. 2018;6:e10799.

Download references

Acknowledgements

We have no acknowledgements to make.

Open access funding provided by Swiss Federal Institute of Technology Zurich. This work was supported by grants from the National Institute for Health and Care Research (COV-LT2-0010) and the funder had no role in the conceptualisation, design, data collection, analysis, decision to publish, or preparation of the manuscript.

Author information

Authors and affiliations.

Sport and Physical Activity Research Institute, School of Health and Life Sciences, University of the West of Scotland, Glasgow, UK

Nilihan E. M. Sanal-Hayes, Marie Mclaughlin, Lawrence D. Hayes, David Carless, Rachel Meach & Nicholas F. Sculthorpe

Future Health Technologies, Singapore-ETH Centre, Campus for Research Excellence and Technological Enterprise (CREATE), Singapore, Singapore

Jacqueline L. Mair

Saw Swee Hock School of Public Health, National University of Singapore, Singapore, Singapore

Long COVID Scotland, 12 Kemnay Place, Aberdeen, UK

Jane Ormerod

Physios for ME, London, UK

Natalie Hilliard

School of Education and Social Sciences, University of the West of Scotland, Glasgow, UK

Joanne Ingram

School of Health and Society, University of Salford, Salford, UK

Nilihan E. M. Sanal-Hayes

School of Sport, Exercise & Rehabilitation Sciences, University of Hull, Hull, UK

Marie Mclaughlin

You can also search for this author in PubMed   Google Scholar

Contributions

Authors’ contributions are given according to the CRediT taxonomy as follows: Conceptualization, N.E.M.S–H., M.M., L.D.H, and N.F.S.; methodology, N.E.M.S–H., M.M., L.D.H., and N.F.S.; software, N.E.M.S–H., M.M., L.D.H., and N.F.S.B.; validation, N.E.M.S–H., M.M., L.D.H, and N.F.S.; formal analysis, N.E.M.S–H., M.M., L.D.H., and N.F.S.; investigation, N.E.M.S–H., M.M., L.D.H., and N.F.S.; resources, L.D.H., J.O., D.C., N.H., J.L.M., and N.F.S.; data curation, N.E.M.S.-H., M.M., L.D.H., and N.F.S.; writing—original draft preparation, N.E.M.S.-H., M.M., L.D.H., and N.F.S.; writing—review and editing, N.E.M.S–H., M.M., L.D.H., J.O., D.C., N.H., R.M., J.L.M., J.I., and N.F.S.; visualisation, N.E.M.S–H. and M.M., supervision, N.F.S; project administration, N.E.M.S–H., M.M., L.D.H., and N.F.S.; funding acquisition, L.D.H., J.O., D.C., N.H., J.L.M., J.I., and N.F.S. All authors have read and agreed to the published version of the manuscript.

Corresponding author

Correspondence to Jacqueline L. Mair .

Ethics declarations

Ethical approval and content to participate.

This manuscript did not involve human participants, data, or tissues, so did not require ethical approval.

Consent for publication

This paper does not contain any individual person’s data in any form.

Competing interests

We report no financial and non-financial competing interests.

Additional information

Publisher's note.

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Supplementary Information

Additional file 1..

Supplementary file 1. Full search string for databse searching.

Rights and permissions

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ . The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/ ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

Reprints and permissions

About this article

Cite this article.

Sanal-Hayes, N.E.M., Mclaughlin, M., Hayes, L.D. et al. A scoping review of ‘Pacing’ for management of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS): lessons learned for the long COVID pandemic. J Transl Med 21 , 720 (2023). https://doi.org/10.1186/s12967-023-04587-5

Download citation

Received : 30 June 2023

Accepted : 03 October 2023

Published : 14 October 2023

DOI : https://doi.org/10.1186/s12967-023-04587-5

Share this article

Anyone you share the following link with will be able to read this content:

Sorry, a shareable link is not currently available for this article.

Provided by the Springer Nature SharedIt content-sharing initiative

• Myalgic encephalomyelitis
• Chronic fatigue syndrome
• Post-exertional malaise

Journal of Translational Medicine

ISSN: 1479-5876

• Submission enquiries: Access here and click Contact Us
• General enquiries: [email protected]

Thank you for visiting nature.com. You are using a browser version with limited support for CSS. To obtain the best experience, we recommend you use a more up to date browser (or turn off compatibility mode in Internet Explorer). In the meantime, to ensure continued support, we are displaying the site without styles and JavaScript.

• View all journals
• Explore content
• About the journal
• Publish with us
• Sign up for alerts
• Published: 15 April 2024

Impact of war on oral health: a systematic review

• Manali Deb Barma 1 ,
• Upendra Singh Bhadauria 2 ,
• Bharathi Purohit 2 ,
• Sneha Malhotra 3 ,
• Deepali Agarwal 2 &
• Harsh Priya 4  

Evidence-Based Dentistry ( 2024 ) Cite this article

1 Altmetric

Metrics details

• Health care
• Periodontitis

To systematically review the evidence from published literature about the impact of war on oral health among children and adults.

Methodology

The electronic search was carried out in the following databases Pubmed, Cochrane, Scopus, Embase, Google Scholar. In vitro studies, cross-sectional, case control studies, and randomized controlled trials conducted during the war time were included. Cross-sectional and case control studies were assessed based on the Newcastle-Ottawa Scale, whereas randomized controlled trials were assessed based on the ROBINS-I tool. In-vitro studies included in this review were assessed using the United States national toxicology program tool.

The search strategy yielded a total of 5126 articles and finally 21 articles were included in the review. The influence of war on oral health was categorized among both the children and adult population. A total of 14 studies were carried out in children whereas the remaining seven studies were carried out in adults. A decrease in dental caries was observed in more war-prone areas due to the limited availability of carbohydrates and sugar. Severe periodontal disease in the adult population, mainly due to less availability of food and diminished oral hygiene habits, was also reported.

Conclusions

This review underscores the multifaceted impact of war on oral health, revealing a notable prevalence of periodontal problems, craniofacial injuries and varying rates of dental caries across affected populations.

This is a preview of subscription content, access via your institution

Access options

Subscribe to this journal

Receive 4 print issues and online access

251,40 € per year

only 62,85 € per issue

Buy this article

• Purchase on Springer Link
• Instant access to full article PDF

Prices may be subject to local taxes which are calculated during checkout

Data availability

The data are available from the corresponding author on reasonable request.

World Health Organization. 2001. The World health report: 2001: Mental health: new understanding, new hope. World Health Organization. https://iris.who.int/handle/10665/42390 .

Murthy RS, Lakshminarayana R. Mental health consequences of war: a brief review of research findings. World Psychiatry. 2006;5:25–30.

PubMed   PubMed Central   Google Scholar  

Summerfield D. War and mental health: a brief overview. Br Med J. 2000;321:232–5. https://doi.org/10.1136/bmj.321.7255.232 .

Article   CAS   Google Scholar  

Mosleh M, Dalal K, Aljeesh Y, Svanström L. The burden of war-injury in the Palestinian health care sector in Gaza Strip. BMC Int Health Hum Rights 2018;18:28.

Article   PubMed   PubMed Central   Google Scholar  

Glick M, et al. A new definition for oral health developed by the FDI World Dental Federation opens the door to a universal definition of oral health. Int Dent J. 2016;66:322–324.

Article   PubMed   Google Scholar  

Norris JM, et al. A modified Newcastle-Ottawa scale for assessment of study quality in genetic urological research. Eur Urol. 2021;79:325–326.

Schünemann HJ, Shea B, Shrier I, Tugwell P, Turner L, Valentine JC, et al. ROBINS-I: a tool for assessing risk of bias in non-randomised studies of interventions. Br Med J. 2016;355:i4919.

Google Scholar  

Ntp. Handbook for conducting a literature-based health assessment using OHAT approach for systematic review and evidence integration. U.S. Dept. of Health and Human Services, National Toxicology Program; 2015.

Hamid SH, Dashash MAD. The impact of post-traumatic stress disorder on dental and gingival status of children during syrian crisis: a preliminary study. J Investig Clin Dent. 2019;10:e12372.

Alhaffar BA, Alawabdi R, Barakat L, Kouchaji C. Oral health and socio-economic status among children during Syrian crisis: a cross-sectional study. BMC Oral Health. 2019;19:165 https://doi.org/10.1186/s12903-019-0856-8 .

Ballouk MA, Dashash M. Caries prevalence and dental health of 8-12 year-old children in Damascus city in Syria during the Syrian crisis; a cross-sectional epidemiological oral health survey. BMC Oral Health. 2019;19:16 https://doi.org/10.1186/s12903-019-0713-9 .

Hamid S, Dashash M, Latifeh Y. A short-term approach for promoting oral health of internally displaced children with PTSD: the key is improving mental health-results from a quasi-randomized trial. BMC Oral Health. 2021;21:58 https://doi.org/10.1186/s12903-020-01385-z .

Riatto SG, Montero J, Pérez DR, Castaño-Séiquer A, Dib A. Oral Health Status of Syrian Children in the Refugee Center of Melilla, Spain. Int J Dent. 2018;2018:2637508 https://doi.org/10.1155/2018/2637508 .

Aldaghir OM, Aljumaily HA, Al-Khannaq YR. War in the Middle East and the Incidence of Cleft Lip and Palate. J Craniofac Surg. 2020;31:e486–e488. https://doi.org/10.1097/SCS.0000000000006495 .

Savabieasfahani M, Ali SS, Bacho R, Savabi O, Alsabbak M. Prenatal metal exposure in the Middle East: imprint of war in deciduous teeth of children. Environ Monit Assess. 2016;188:505 https://doi.org/10.1007/s10661-016-5491-0 .

Article   CAS   PubMed   PubMed Central   Google Scholar  

Jahanbin A, Kianifar H, Yaghoubi-Al Z, Malekian A, Keikhaee B, Hasanzadeh N, et al. Had prevalence of cleft lip and palate differed during the Iran-Iraq war? J Craniofac Surg. 2013;24:826–829. https://doi.org/10.1097/SCS.0b013e318290273d .

Ahmed NA, Astrøm AN, Skaug N, Petersen PE. Dental caries prevalence and risk factors among 12-year old schoolchildren from Baghdad, Iraq: a post-war survey. Int Dent J. 2007;57:36–44. https://doi.org/10.1111/j.1875-595x.2007.tb00116.x .

Yassaei S, Rafieian M, Ghafari R. Abnormal oral habits in the children of war veterans. J ClinPediatr Dent. 2005;29:189–192. https://doi.org/10.17796/jcpd.29.3.08r72m4580852h84 .

Rajić Z, Radionov D, Rajić-Mestrović S. Trends in dental caries in 12-year old children in Croatia. CollAntropol. 2000;24:21–24.

TOVERUD G. Decrease in caries frequency in Norwegian children during World War II. J Am Dent Assoc. 1949;39:128–136.

Article   CAS   PubMed   Google Scholar  

Arheiam AA, Elareibi I, Elatrash A, Baker SR. Prevalence and factors associated with traumatic dental injuries among schoolchildren in war-torn Libya. Dent Traumatol. 2020;36:185–191. https://doi.org/10.1111/edt.12529 .

Arheiam AA, Harris RV, Baker SR. Changes in dental caries and sugar intake before and during the conflict in Libya: a natural experiment. Community Dent Oral Epidemiol. 2020;48:201–207. https://doi.org/10.1111/cdoe.12526 .

Suman M, Spalj S, Plancak D, Dukic W, Juric H. The influence of war on the oral health of professional soldiers. Int Dent J. 2008;58:71–74. https://doi.org/10.1111/j.1875-595x.2008.tb00178.x .

Muhvić-Urek M, Uhac I, Vuksić-Mihaljević Z, Leović D, Blecić N, Kovac Z. Oral health status in war veterans with post-traumatic stress disorder. J Oral Rehabil. 2007;34:1–8. https://doi.org/10.1111/j.1365-2842.2006.01674.x .

Kazwini R, Kasem T, Alhuda NEA, Albarshah M, Subeh D, Alhaffar MHDBA. Oral health assessment in a sample of displaced people as a result of the war in Syria in Damascus city: results of non-profit initiative. BMC Oral Health. 2021;21:532 https://doi.org/10.1186/s12903-021-01874-9 .

Mottaghi A, Hoseinzade A, Zamani E, Araghizade HA. Status of dental health in chemical warfare victims: the case of Isfahan, Iran. Indian J Dent Res. 2012;23:506–508. https://doi.org/10.4103/0970-9290.104959 .

Ivanković A, Lukić IK, Ivanković Z, Radić A, Vukić I, Simić A. Dental caries in postwar Bosnia and Herzegovina. Community Dent Oral Epidemiol. 2003;31:100–104. https://doi.org/10.1034/j.1600-0528.2003.00052.x .

Levin L, Zadik Y, Peleg K, Bigman G, Givon A, Lin S. Incidence and severity of maxillofacial injuries during the Second Lebanon War among Israeli soldiers and civilians. J Oral Maxillofac Surg. 2008;66:1630–1633. https://doi.org/10.1016/j.joms.2007.11.028 .

Chan RK, Siller-Jackson A, Verrett AJ, Wu J, Hale RG. Ten years of war: a characterization of craniomaxillofacial injuries incurred during operations Enduring Freedom and Iraqi Freedom. J Trauma Acute Care Surg. 2012;73:S453–S458. https://doi.org/10.1097/TA.0b013e3182754868 .

de Abreu MHNG, Cruz AJS, Borges-Oliveira AC, Martins RC, Mattos FF. Perspectives on social and environmental determinants of oral health. Int J Environ Res Public Health. 2021;18:13429 https://doi.org/10.3390/ijerph182413429 .

Torppa-Saarinen E, Tolvanen M, Lahti S, Suominen AL. Changes and determinants of unmet oral health treatment need. Community Dent Oral Epidemiol. 2021;49:158–165. https://doi.org/10.1111/cdoe.12587 .

Ghanbarzadegan A, Balasubramanian M, Luzzi L, Brennan D, Bastani P. Inequality in dental services: a scoping review on the role of access toward achieving universal health coverage in oral health. BMC Oral Health. 2021;21:404 https://doi.org/10.1186/s12903-021-01765-z .

Download references

Author information

Authors and affiliations.

Agartala Government Dental College & IGM Hospital, Agartala, India

Manali Deb Barma

Division of Public Health Dentistry, CDER-AIIMS, New Delhi, India

Upendra Singh Bhadauria, Bharathi Purohit & Deepali Agarwal

Maulana Azad Institute of Dental Sciences, New Delhi, India

Sneha Malhotra

Division of Public Health Dentistry, Centre for Dental Education and Research, AIIMS, New Delhi, India

Harsh Priya

You can also search for this author in PubMed   Google Scholar

Contributions

MDB: conception, data acquisition and drafted the manuscript. USB: Data acquisition, visualization, interpretation, and drafted the manuscript. BP: synthesis and critically revised the paper. SM: Data acquisition and synthesis. DA: Data acquisition and interpretation. HP: Conception, provision and management of study literature resources, critically revised the paper. All authors gave their final approval and agreed to be accountable for all aspects of the work. The authors read and approved the final paper. Author 1 and 2 have contributed equally.

Corresponding author

Correspondence to Harsh Priya .

Ethics declarations

Competing interests.

The authors declare no competing interests.

Additional information

Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Supplementary information

Supplementary information, rights and permissions.

Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Cite this article.

Barma, M.D., Bhadauria, U.S., Purohit, B. et al. Impact of war on oral health: a systematic review. Evid Based Dent (2024). https://doi.org/10.1038/s41432-024-01006-6

Download citation

Received : 13 February 2024

Accepted : 03 April 2024

Published : 15 April 2024

DOI : https://doi.org/10.1038/s41432-024-01006-6

Share this article

Anyone you share the following link with will be able to read this content:

Sorry, a shareable link is not currently available for this article.

Provided by the Springer Nature SharedIt content-sharing initiative

Quick links

• Explore articles by subject
• Guide to authors
• Editorial policies

• Open access
• Published: 13 April 2024

Associations between transport modes and site-specific cancers: a systematic review and meta-analysis

• Win Thu 1 ,
• Alistair Woodward 1 ,
• Alana Cavadino 1 &
• Sandar Tin Tin 1 , 2  

Environmental Health volume  23 , Article number:  39 ( 2024 ) Cite this article

90 Accesses

Metrics details

Physical inactivity is a global public health problem. A practical solution would be to build physical activity into the daily routine by using active modes of transport. Choice of transport mode can influence cancer risk through their effects on levels of physical activity, sedentary time, and environmental pollution. This review synthesizes existing evidence on the associations of specific transport modes with risks of site-specific cancers.

Relevant literature was searched in PubMed, Embase, and Scopus from 1914 to 17th February 2023. For cancer sites with effect measures available for a specific transport mode from two or more studies, random effects meta-analyses were performed to pool relative risks (RR) comparing the highest vs. lowest activity group as well as per 10 Metabolic Equivalent of Task (MET) hour increment in transport-related physical activity per week ( ∼ 150 min of walking or 90 min of cycling).

27 eligible studies (11 cohort, 15 case-control, and 1 case-cohort) were identified, which reported the associations of transport modes with 10 site-specific cancers. In the meta-analysis, 10 MET hour increment in transport-related physical activity per week was associated with a reduction in risk for endometrial cancer (RR: 0.91, 95% CI: 0.83–0.997), colorectal cancer (RR: 0.95, 95% CI: 0.91–0.99) and breast cancer (RR: 0.99, 95% CI: 0.89–0.996). The highest level of walking only or walking and cycling combined modes, compared to the lowest level, were significantly associated with a 12% and 30% reduced risk of breast and endometrial cancers respectively. Cycling, compared to motorized modes, was associated with a lower risk of overall cancer incidence and mortality.

Active transport appears to reduce cancer risk, but evidence for cancer sites other than colorectum, breast, and endometrium is currently limited.

Peer Review reports

Introduction

Physical inactivity is a global public health problem, contributing to substantial disease and economic burden worldwide [ 1 , 2 ]. With rapid changes in technology, lifestyle, and habitual environment, people have been less active and more sedentary over the past few decades. Globally, about 1 in 4 adults were not active, i.e., did not meet the World Health Organization (WHO) recommendation of engaging at least 150–300 min of moderate-intensity or 75–150 min of vigorous-intensity aerobic physical activity per week [ 3 ], but the prevalence varied widely within and across countries [ 4 ]. If the current trends continue, it is unlikely that the WHO’s target to reduce physical inactivity by 10% in 2025 will be met.

One practical solution would be to build physical activity into the daily routine by using active modes of transport [ 5 ]. Walking and cycling have been shown to improve health (mainly all-cause mortality, cardiovascular disease, diabetes, and cancer) [ 6 ] and also provide social, economic and environmental benefits [ 7 , 8 ]. Car use, on the other hand, contributes to a significant proportion of daily sedentary time, and the situation is worsening with increasing traffic congestion/delays [ 9 ]; it has been associated with an increased risk of obesity and related outcomes [ 10 ]. Further, exposure to environmental pollutants such as nitrogen dioxide and/or particulate matter could differ across different road users [ 11 ], while it has been shown to increase the risk of certain cancers, particularly lung cancer [ 12 ].

While there is ample evidence linking leisure time physical activity or physical activity in general with a reduced risk for a number of cancer sites [ 13 , 14 ], and sedentary behavior in general with an increased risk [ 15 ], the findings may not be directly applicable to transport-related activity because the context and correlates of activity as well as its frequency, duration and intensity are likely to be different across different domains. We therefore reviewed the existing literature that reported the associations between transport modes and risks of site-specific cancers.

A systematic literature review and meta-analysis was conducted and reported according to the PRISMA guideline (Supplementary file S1 ). The review was not registered.

Search strategy and study selection

Relevant literature was searched from 1914 to 17th February 2023 in PubMed, Scopus, and Embase databases using the relevant search terms such as walking, cycling, car, public transport, commute and cancers. Site-specific cancers known to be associated with physical activity and body weight such as breast, colon, liver, esophageal adenocarcinoma and those associated with environmental factor such as lung and melanoma of skin were also searched (Supplementary file S2 ). The reference lists of systematic reviews on physical activity and cancers were also reviewed. Studies were included if they (1) used cohort, case-control, case-cohort or experimental design, (2) assessed transport modes such as walking, cycling, public transport or car use as the exposures of interest, (3) investigated one or more site-specific cancers, overall cancer incidence and/or mortality as the outcome(s), (4) reported effect measures associated with transport modes, and (5) published the full article in English. Studies that used cross-sectional design or mathematical modeling to estimate health impacts at the population level were excluded. Details of excluded studies after full text review, together with the reasons for exclusion, were provided in the Supplementary file S3 . WT conducted the search and selection, and STT oversaw the process.

Data extraction and study quality assessment

Information about title, first author, year of publication, study name (if available), country, study design, sample size, age range of the participants, follow-up duration (for cohort and case-cohort studies), data collection tool, measurement units for exposure(s), data sources for outcome(s), site-specific cancer assessed, effect measures, and confounders adjusted were extracted in a standardized data collection spreadsheet. The study quality was evaluated using the Newcastle-Ottawa Scale (NOS) [ 16 ], which scores the cohort and case-control studies based on three domains: selection of study groups, comparability of the groups and ascertainment of exposure (case-control studies) or outcome (cohort studies). For the second domain, a point was awarded for adjustment of Body Mass Index (BMI) - to evaluate the direct vs. indirect (through BMI) effect of physical activity on cancer risk, and another point for adjustment of physical activities from other domains - to isolate the effects of transport-related physical activity from other activities. A maximum of nine points were awarded, with a higher score indicating better quality [ 16 ]. For case-cohort studies, the NOS scale for cohort studies was used. WT conducted the data extraction and quality assessment, and STT oversaw the process.

Data analysis

For cancer sites with effect measures available for a specific transport mode from two or more studies, meta-analyses were performed using random effects models. The analyses compared the highest level of active transport such as walking, cycling or mixed mode with the lowest level as reported in the individual studies. Where necessary, the reference category for exposure was changed to the lowest group to facilitate pooling of the risks [ 17 ]. The pooled relative risks (RRs) and 95% CI were presented for breast, endometrial, colorectal and testicular cancers, and overall cancer mortality.

For studies that reported time or MET as measurement units, the dose-response effects were estimated using the trend estimation method proposed by Greenland and Longnecker [ 18 ]. The reported time spent for each mode/category was converted to MET hours (see Supplementary file S4 for conversion values and formulas used). For studies that only reported estimates for categorical exposures, study-specific slopes were calculated from the natural logs of the reported risk estimates across categories and risk estimates per unit change were then estimated. The pooled results were presented per 10 Metabolic Equivalent of Task (MET) hour increment in transport-related physical activity per week ( ∼ 150 min of walking or 90 min of cycling) to align with the WHO’s physical activity recommendation [ 3 ]. This approach enabled us to pool risk estimates from a large number of studies irrespective of how the exposures were assessed (e.g., walking and cycling separately or combined) or categorised. The results were presented for breast, endometrial, colorectal, prostate cancers, and overall cancer mortality.

Meta-analysis was not conducted for the studies that compared active and non-active modes in relation to overall cancer incidence and mortality due to the potential overlap of the study samples.

For meta-analyses involving four or more studies, publication bias was assessed through the visual inspection of funnel plots, Begg’s rank correlation test, and Egger’s regression test for asymmetry. If significant associations were observed, sensitivity analyses were conducted by removing one study at a time from the initial meta-analysis to test the robustness of the results. Where possible, sub-group analyses were performed to assess variability of summary effects across population groups (Western vs. Asian), study design (cohort vs. case-control), measurement units (time vs. MET) and adjustment for BMI (yes vs. no). Metafor [ 19 ] and dosresmeta [ 20 ] R packages were used for meta-analysis and trend estimation. All authors have access to the data.

Of the 11,829 records identified, 27 unique studies (total 34 publications) were included, of which 22 studies (28 records) contributed to the meta-analyses (Fig.  1 ). There were four publications from the Netherlands Cohort Study which reported endometrial [ 21 ], ovarian [ 22 ], prostate [ 23 ], and colorectal [ 24 ] cancers, three publications from United Kingdom Biobank which reported lung [ 25 ], breast and colon [ 26 ], and overall cancer incidence and mortality [ 27 ], two publications from Shanghai Women’s Health Study which reported breast [ 28 ] and overall cancer mortality [ 29 ], and two publications from National Institutes of Health - American Association of Retired Persons Diet and Health Study which reported breast [ 30 ] and endometrial [ 31 ] cancers. Of the included studies, 20 compared the risks between the highest and lowest levels of active transport (e.g., walking, cycling, walking and cycling) and two compared the risk between active and non-active commuting modes. The majority used case-control design ( n  = 15), followed by cohort ( n  = 11) and case-cohort ( n  = 1) designs. Most of the studies were conducted in North America, mainly in the United States (US) ( n  = 7), followed by Europe ( n  = 5), China ( n  = 5), United Kingdom ( n  = 4), Australia ( n  = 2) and the remaining four studies were from India, Iran, Brazil and Nigeria. (Table  1 )

Almost half of the studies assessed walking and cycling combined, i.e., did not provide the risk estimates for each mode ( n  = 13), while others assessed walking and cycling separately ( n  = 8), or assessed only one mode (walking: n  = 3 and cycling: n  = 3). Most studies quantified active transport in terms of time spent (e.g., minutes per day, hours per week) ( n  = 14) or MET ( n  = 7), but others assessed it in terms of activity status (e.g., yes, no) ( n  = 3), or in comparison to car or motorized mode ( n  = 2), and distance ( n  = 1) (Supplementary file S5 ). The studies reported the risks associated with ten site-specific cancers, most commonly breast ( n  = 12), endometrial ( n  = 5), and colorectal ( n  = 4) cancers (Fig.  2 ). Cancer cases were identified through cancer registries, death registries, pathological reports, or hospital or medical records (Supplementary file S5 ). The NOS score for cohort studies ranged from 5 to 9, with an average score of 6.5, and the score for case-control studies ranged from 4 to 7, with an average score of 5.6 (Table  1 , detailed scoring in Supplementary file S6 , S7 ).

Flow diagram for study selection

Cancers reported in the studies

Active transport studies

The pooled results were presented for breast, endometrial, colorectal, testicular and prostate cancers, and overall cancer mortality (Fig.  3 ). For other cancers where only one study was identified, the results from the individual study were presented.

Breast cancer

In the meta-analysis of six studies comparing the highest vs. lowest activity group, an inverse association was observed for walking (RR: 0.88, 95% CI: 0.78–0.98), a borderline inverse association for cycling (RR: 0.90, 95% CI: 0.77–1.05) and no significant association for walking and cycling combined (RR: 0.97, 95% CI: 0.84–1.12). 10 MET hour increment in transport-related physical activity per week ( ∼ 150 min of walking or 90 min of cycling) was associated with a marginally reduced risk (RR: 0.99, 95% CI: 0.97–0.996). (Fig. 3 , detailed forest plots in the supplementary file S9 )

Endometrial cancer

The meta-analysis of four studies indicated that walking and cycling combined was associated with a reduced risk of endometrial cancer (RR comparing highest vs. lowest: 0.70, 95% CI: 0.56–0.87; RR per 10 MET hour increment in activity per week: 0.91, 95% CI: 0.83–0.997). (Fig.  3 , detailed forest plots in S9 )

Colorectal cancer

In the meta-analysis of two studies, walking and cycling combined was associated with a reduced risk of colorectal cancer (RR comparing highest vs. lowest: 0.89, 95% CI: 0.78–1.01; RR per 10 MET hour increment in activity per week: 0.95, 95% CI: 0.91–0.99) (Fig.  3 , detailed forest plot in S9 ).

Testicular cancer

In the meta-analysis of two studies, there was no significant association between cycle commuting in adolescence and testicular germ cell cancer (RR comparing highest vs. lowest: 1.23, 95% CI: 0.71–2.13). (Fig.  3 , detailed forest plot in S9 )

Prostate cancer

10 MET hour increment per week for transport related physical activity was associated with a reduced risk of prostate cancer (RR: 0.96, 95% CI: 0.88–1.04) (Fig.  3 , detailed forest plot in S9 ).

Ovarian cancer

Only a case-cohort study assessed the relationship of walking and cycling combined mode with ovarian cancer risk, and reported no significant association (Supplementary file S5 ) [ 22 ].

Liver, gallbladder and biliary tract cancers

A cohort study reported a significant association of commuting physical activity with a reduced risk of gallbladder and biliary tract cancers in women (HR: 0.51, 95% CI: 0.28–0.94) but not in men (HR: 0.92, 95% CI: 0.61–1.37); there was no significant association with liver cancer in both sexes (supplementary file S5 ) [ 49 ].

Renal cancer

A case-control study assessed the association of walking and cycling with risk of renal cell carcinoma in white and black participants in the ages of 20s and 50s, and reported a significant association in the white participants in their 20s (OR comparing lowest vs. highest: 1.42, 95% CI: 1.10–1.83) but not in the black counterparts; the associations were also not significant in both groups in their 50s. (Supplementary file S5 ) [ 50 ].

Overall cancer mortality

In the meta-analyses of two studies, there was an inverse association for cycling only (RR comparing highest vs. lowest: 0.60, 95% CI: 0.34–1.04) and walking and cycling combined (RR: 0.98, 95% CI: 0.86–1.12), and also per 10 MET hour increment in activity per week (RR: 0.97, 95% CI: 0.92–1.01). (Fig.  3 , detailed forest plots in S9 )

In sub-group analyses, similar associations were observed between walking and breast cancer risk in terms of study design (cohort, case-control), population (western vs. Asian), measurement unit (time vs. MET), menopausal status (premenopausal and postmenopausal) and adjustment of BMI (yes vs. no); however, the associations were stronger in studies that adjusted for physical activity from other domains (Supplementary file S10 ). In the leave-one-out analyses assessing walking and breast cancer risk, the results were sensitive to effect sizes from some studies, but this was not the case for walking and cycling combined mode and endometrial cancer (Supplementary file S11 ). There was no evidence for funnel plot asymmetry; Egger’s regression tests and Begg’s ranks correlation tests were not significant (Supplementary file S12 ).

Results of meta-analysis for active transport studies. RE = a random-effects model, MET = Metabolic Equivalent of Task, I 2  = I 2 statistics for heterogeneity, RR = Summary relative risk

Studies comparing active vs. non-active modes for commuting

Four eligible publications were identified, of which three used the data from UK Biobank [ 25 , 26 , 27 ], one used the UK census data [ 51 ]. Three reported the associations for overall cancer incidence and mortality, and one reported the risk associated with lung cancer (Fig.  4 ). In the study that assessed lung cancer using the data from UK Biobank, when compared to automobile only mode, active modes did not show a significant association whereas frequent use of public transport (≥ 5 trips per week) was associated with an increased risk of lung cancer (HR: 1.58, 95% CI: 1.08–2.33) [ 25 ] (Fig.  4 ). In another UK Biobank study, no significant associations were observed for breast and colon cancers, and overall cancer incidence and mortality when more active patterns of commuting (walking, cycling, public transport, either alone or in combination with car) were compared to car only mode [ 26 ].

The results of two studies [ 27 , 51 ] that assessed overall cancer incidence and mortality were not combined as the outcome data was extracted from the same national cancer registry with an overlapped time frame (1991–2011 and 2007–2014), although the exposure information came from different sources (census and UK Biobank). In these studies, compared to private motorized mode or non-active mode, cycling was inversely associated with overall cancer incidence and mortality. Walking and public transport were also inversely associated with overall cancer incidence in the study that used the census data [ 51 ].

Results of the individual studies comparing active vs. non-active modes for commuting. Private motorized mode = car or motorcycle, Non-active = car or public transport, Active patterns of commuting = any other patterns including walking, cycling, public transport, either alone or in combination with car, HR = Hazard Ratio, regular:1–4, often: ≥5 work-bound trips/week

This review identified 27 studies (34 publications) that reported the associations of specific transport modes, mainly active transport modes, with risks of ten site-specific cancers along with overall cancer incidence and mortality. The most frequently studied cancer sites were breast, endometrium, and colorectum; our meta-analysis showed a reduction in risk of these cancers (1%, 9% and 5%, respectively) per 10 MET hour per week increment in transport-related physical activity ( ∼ 150 min of walking or 90 min of cycling).

We found an inverse association between active transport and risks of breast and endometrial cancers, with similar magnitude of risk reduction observed in previous systematic reviews on physical activity in general [ 55 , 56 ]. While obesity is known to increase post-menopausal but not pre-menopausal breast cancer risk [ 57 ], we found similar results by menopausal status. In contrast, an earlier review did not find any significant association between walking in general and risk of pre- or post-menopausal breast cancer [ 58 ], possibly because compared to walking for transport, walking for leisure or at home generally uses lower energy [ 59 ], and therefore may have less effect on body weight.

The inverse association of active transport with colorectal cancer risk observed in this review is also consistent with the findings from existing reviews on transport-related physical activity [ 60 ] as well as physical activity in general [ 61 ]. While physical activity in general or for leisure has also been associated with a reduced risk of many other cancer sites including liver, gastric, renal and lung [ 13 , 14 ], the evidence related to transport-related physical activity is currently limited.

Mechanisms linking physical activity with specific cancer sites have been proposed, including its effects on sex hormones (breast, endometrial and prostate cancers), insulin sensitivity, glucose metabolism and adipokines (obesity-related cancers), and inflammation and immune function (most cancers) [ 62 ]. For colorectal cancer, another potential mechanism is reduced contact time between carcinogens and bowel mucosa cells due to exercise-induced intestinal mobility [ 63 ].

The overall quality of the included studies, evaluated by NOS score, ranged from 4 to 9, and in general, cohort studies tend to have higher scores compared to case-control studies. The common criteria the studies did not meet include: inadequate exposure assessment, loss to follow-up (cohort studies) and low response rates (case-control studies). While we were not able to undertake subgroup analyses by NOS score due to the limited number of studies available, our subgroup analyses by study design showed similar associations between walking and breast cancer in cohort vs. case-control studies.

To our knowledge, this review represents the first systematic attempt to synthesize the existing evidence on specific transport modes and site-specific cancers. We provided mode-specific summary effects where possible and calculated the dose-response effects for transport-related physical activity, in line with WHO physical activity recommendation. When interpreting the findings, some limitations need to be considered. First, the review may not have included some eligible studies published in languages other than English. Second, due to the limited number of available studies, we were not able to pool the results separately for cohort and case-control studies; however, we conducted sub-group analyses by study design where possible. We were not able to evaluate the non-linear relationship between transport-related physical activity and the risks of site-specific cancers. While a recent systematic review on breast and colon cancers reported a linear relationship with physical activity [ 64 ], others suggested a non-linear relationship between physical activity and cancer risk [ 65 , 66 ]. Further, variations in measurement and categorization of the exposure across the studies make direct comparison of the results between different modes (e.g., walking vs. cycling) difficult. Finally, the majority of the studies included were conducted in high income countries in Europe, UK, and North America, limiting the generalizability of the findings to other populations and low and middle income countries where urbanization and motorization are mainly taking place [ 67 ].

Our findings suggest that transport choices may influence cancer risk, particularly of obesity-related cancers such as breast, colon and endometrial cancers. Breast cancer is the most common cancer in women globally, with an estimated over 2 million new cases (11.7% of all new cases) in 2020, while colon cancer stood at fourth place (over 1 million cases, 6% of total cases) [ 68 ]. The incidence of endometrial cancer also seems to be increasing in many countries particularly in younger women. Our findings indicate that the risks of these cancers can be reduced by meeting the WHO physical activity recommendation through active commuting ( ∼ 150 min of walking or 90 min of cycling per week). Yet, the current evidence is limited in relation to other cancer sites, underlying mechanisms, and potential environmental influences, requiring further exploration.

Given heterogeneity in exposure measurements in the existing studies, harmonizing choice of the assessment tool (e.g., using International Physical Activity Questionnaires that can capture information about all four physical activity domains including transport modes), and reporting the dose-response estimates for each transport mode such as walking and cycling separately rather than a combined mode would enhance comparability of results and provide mode-specific effects. Repeated or regular assessments of exposures/transport modes used throughout the study duration would capture changes and their potential impact on outcomes in cohort studies. Importantly, more research is needed in low and middle-income settings to generate context-specific evidence.

In conclusion, active transport modes appear to reduce cancer risk, but evidence for cancer sites other than colorectum, breast and endometrium is currently limited.

Data availability

No datasets were generated or analysed during the current study.

Lee IM, Shiroma EJ, Lobelo F, Puska P, Blair SN, Katzmarzyk PT. Effect of physical inactivity on major non-communicable diseases worldwide: an analysis of burden of disease and life expectancy. Lancet. 2012;380(9838):219–29. https://doi.org/10.1016/S0140-6736(12)61031-9 .

Article   Google Scholar  

Ding D, Lawson KD, Kolbe-Alexander TL, et al. The economic burden of physical inactivity: a global analysis of major non-communicable diseases. Lancet. 2016;388(10051):1311–24. https://doi.org/10.1016/S0140-6736(16)30383-X .

World Health Organization. Global status report on physical activity 2022.; 2022.

Guthold R, Stevens GA, Riley LM, Bull FC. Worldwide trends in insufficient physical activity from 2001 to 2016: a pooled analysis of 358 population-based surveys with 1·9 million participants. Lancet Global Health. 2018;6(10):e1077–86. https://doi.org/10.1016/S2214-109X(18)30357-7 .

Berrigan D, Troiano RP, McNeel T, DiSogra C, Ballard-Barbash R. Active transportation increases adherence to activity recommendations. Am J Prev Med. 2006;31(3):210–6. https://doi.org/10.1016/j.amepre.2006.04.007 .

Dinu M, Pagliai G, Macchi C, Sofi F. Active commuting and multiple Health outcomes: a systematic review and Meta-analysis. Sports Med. 2019;49(3):437–52. https://doi.org/10.1007/s40279-018-1023-0 .

Boniface S, Scantlebury R, Watkins SJ, Mindell JS. Health implications of transport: evidence of effects of transport on social interactions. J Transp Health. 2015;2(3):441–6. https://doi.org/10.1016/j.jth.2015.05.005 .

Higgins PAT. Exercise-based transportation reduces oil dependence, carbon emissions and obesity. Envir Conserv. 2005;32(3):197–202. https://doi.org/10.1017/S037689290500247X .

Article   CAS   Google Scholar  

World Health Organization. Global Status Report on Road Safety 2018: Summary.; 2018.

Sugiyama T, Chandrabose M, Homer AR, Sugiyama M, Dunstan DW, Owen N. Car use and cardiovascular disease risk: systematic review and implications for transport research. J Transp Health. 2020;19:100930. https://doi.org/10.1016/j.jth.2020.100930 .

Panchal R, Panagi M, May HR, et al. Personal air pollution exposure during morning commute car and active transport journeys. J Transp Health. 2022;26:101365. https://doi.org/10.1016/j.jth.2022.101365 .

Turner MC, Andersen ZJ, Baccarelli A, et al. Outdoor air pollution and cancer: an overview of the current evidence and public health recommendations. CA Cancer J Clin. 2020;70(6):460–79. https://doi.org/10.3322/caac.21632 .

Moore SC, Lee IM, Weiderpass E, et al. Association of leisure-time physical activity with risk of 26 types of Cancer in 1.44 million adults. JAMA Intern Med. 2016;176(6):816. https://doi.org/10.1001/jamainternmed.2016.1548 .

Rezende LFMD, Sá THD, Markozannes G, et al. Physical activity and cancer: an umbrella review of the literature including 22 major anatomical sites and 770 000 cancer cases. Br J Sports Med. 2018;52(13):826–33. https://doi.org/10.1136/bjsports-2017-098391 .

Hermelink R, Leitzmann MF, Markozannes G, et al. Sedentary behavior and cancer–an umbrella review and meta-analysis. Eur J Epidemiol. 2022;37(5):447–60. https://doi.org/10.1007/s10654-022-00873-6 .

Wells G, Shea B, O’Connell D et al. The Newcastle-Ottawa Scale (NOS) for assessing the quality of nonrandomised studies in meta-analyses. The Newcastle-Ottawa Scale (NOS) for assessing the quality of nonrandomised studies in meta-analyses. https://www.ohri.ca/programs/clinical_epidemiology/oxford.asp .

Taylor K. Wanting a particular reference category in categorical risk data. Data extraction tips for meta-analysis. https://www.cebm.ox.ac.uk/resources/data-extraction-tips-meta-analysis/reference-category-risk-data .

Greenland S, Longnecker MP. Methods for Trend Estimation from Summarized Dose-Response Data, with applications to Meta-Analysis. Am J Epidemiol. 1992;135(11):1301–9. https://doi.org/10.1093/oxfordjournals.aje.a116237 .

Viechtbauer W. Conducting Meta-analyses in R with the metafor Package. J Stat Soft. 2010;36(3). https://doi.org/10.18637/jss.v036.i03 .

Crippa A, Orsini N. Multivariate Dose-Response Meta-Analysis: The dosresmeta R Package. J Stat Soft. 2016;72(Code Snippet 1). https://doi.org/10.18637/jss.v072.c01 .

Schouten LJ, Goldbohm RA, van den Brandt PA, Anthropometry. Physical activity, and Endometrial Cancer Risk: results from the Netherlands Cohort Study. JNCI J Natl Cancer Inst. 2004;96(21):1635–8. https://doi.org/10.1093/jnci/djh291 .

Biesma RG, Schouten LJ, Dirx MJM, Goldbohm RA, van den Brandt PA. Physical activity and risk of ovarian Cancer: results from the Netherlands Cohort Study (the Netherlands). Cancer Causes Control. 2006;17(1):109–15. https://doi.org/10.1007/s10552-005-0422-3 .

Zeegers MPA, Dirx MJM, van den Brandt PA. Physical activity and the risk of prostate Cancer in the Netherlands Cohort Study, results after 9.3 years of follow-up. Cancer Epidemiol Biomarkers Prev. 2005;14(6):1490–5. https://doi.org/10.1158/1055-9965.EPI-04-0771 .

Simons CCJM, Hughes LAE, van Engeland M, Goldbohm RA, van den Brandt PA, Weijenberg MP. Physical activity, occupational sitting time, and Colorectal Cancer Risk in the Netherlands Cohort Study. Am J Epidemiol. 2013;177(6):514–30. https://doi.org/10.1093/aje/kws280 .

Wong JYY, Jones RR, Breeze C, et al. Commute patterns, residential traffic-related air pollution, and lung cancer risk in the prospective UK Biobank cohort study. Environ Int. 2021;155:106698. https://doi.org/10.1016/j.envint.2021.106698 .

Panter J, Mytton O, Sharp S, et al. Using alternatives to the car and risk of all-cause, cardiovascular and cancer mortality. Heart. 2018;104(21):1749–55. https://doi.org/10.1136/heartjnl-2017-312699 .

Celis-Morales CA, Lyall DM, Welsh P, et al. Association between active commuting and incident cardiovascular disease, cancer, and mortality: prospective cohort study. BMJ Published Online April. 2017;19:j1456. https://doi.org/10.1136/bmj.j1456 .

Pronk A, Ji BT, Shu XO, et al. Physical activity and breast cancer risk in Chinese women. Br J Cancer. 2011;105(9):1443–50. https://doi.org/10.1038/bjc.2011.370 .

Matthews CE, Jurj AL, Shu Xo, et al. Influence of Exercise, walking, Cycling, and overall nonexercise physical activity on Mortality in Chinese women. Am J Epidemiol. 2007;165(12):1343–50. https://doi.org/10.1093/aje/kwm088 .

George SM, Irwin ML, Matthews CE, et al. Beyond recreational physical activity: examining Occupational and Household Activity, Transportation Activity, and sedentary behavior in relation to postmenopausal breast Cancer Risk. Am J Public Health. 2010;100(11):2288–95. https://doi.org/10.2105/AJPH.2009.180828 .

Gierach GL, Chang SC, Brinton LA, et al. Physical activity, sedentary behavior, and endometrial cancer risk in the NIH-AARP Diet and Health Study. Int J Cancer. 2009;124(9):2139–47. https://doi.org/10.1002/ijc.24059 .

Luoto R, Latikka P, Pukkala E, Hakulinen T, Vihko V. The effect of physical activity on breast cancer risk: a cohort study of 30,548 women. Eur J Epidemiol. 2000;16(10):973–80. https://doi.org/10.1023/A:1010847311422 .

Gomes MLB, Pinto SS, Domingues MR. Physical activity and breast Cancer: a case-control study in Southern Brazil. Nutr Cancer. 2022;74(1):149–57. https://doi.org/10.1080/01635581.2021.1880607 .

Azubuike SO, Hayes L, Sharp L, Alabi A, Oyesegun RA, McNally R. Physical activity and the risk of breast cancer among Nigerian women. Cancer Epidemiol. 2022;78:102163. https://doi.org/10.1016/j.canep.2022.102163 .

Si S, Boyle T, Heyworth J, Glass DC, Saunders C, Fritschi L. Lifetime physical activity and risk of breast cancer in pre-and post-menopausal women. Breast Cancer Res Treat. 2015;152(2):449–62. https://doi.org/10.1007/s10549-015-3489-x .

Mathew A, Gajalakshmi V, Rajan B, et al. Physical activity levels among urban and rural women in south India and the risk of breast cancer: a case–control study. Eur J Cancer Prev. 2009;18(5):368–76. https://doi.org/10.1097/CEJ.0b013e32832e1c46 .

Steindorf K. Case-control study of physical activity and breast Cancer risk among Premenopausal women in Germany. Am J Epidemiol. 2003;157(2):121–30. https://doi.org/10.1093/aje/kwf181 .

John EM, Horn-Ross PL, Koo J. Lifetime physical activity and breast cancer risk in a multiethnic population: the San Francisco Bay area breast cancer study. Cancer Epidemiol Biomarkers Prev. 2003;12(11 Pt 1):1143–52.

Google Scholar  

Matthews CE, Shu XO, Jin F, et al. Lifetime physical activity and breast cancer risk in the Shanghai breast Cancer Study. Br J Cancer. 2001;84(7):994–1001. https://doi.org/10.1054/bjoc.2000.1671 .

Marcus PM, Newman B, Moorman PG, et al. Physical activity at age 12 and adult breast cancer risk (United States). Cancer Causes Control. 1999;10(4):293–302. https://doi.org/10.1023/A:1008971417282 .

Friberg E, Mantzoros CS, Wolk A. Physical activity and risk of Endometrial Cancer: a Population-based prospective cohort study. Cancer Epidemiol Biomarkers Prev. 2006;15(11):2136–40. https://doi.org/10.1158/1055-9965.EPI-06-0465 .

John EM, Koo J, Horn-Ross PL. Lifetime physical activity and risk of Endometrial Cancer. Cancer Epidemiol Biomarkers Prev. 2010;19(5):1276–83. https://doi.org/10.1158/1055-9965.EPI-09-1316 .

Matthews CE, Xu WH, Zheng W, et al. Physical activity and risk of Endometrial Cancer: a report from the Shanghai Endometrial Cancer Study. Cancer Epidemiol Biomarkers Prev. 2005;14(4):779–85. https://doi.org/10.1158/1055-9965.EPI-04-0665 .

Mahmood S, English DR, MacInnis RJ, et al. Domain-specific physical activity and the risk of colorectal cancer: results from the Melbourne Collaborative Cohort Study. BMC Cancer. 2018;18(1):1063. https://doi.org/10.1186/s12885-018-4961-x .

Hou L. Commuting physical activity and risk of Colon cancer in Shanghai, China. Am J Epidemiol. 2004;160(9):860–7. https://doi.org/10.1093/aje/kwh301 .

Littman AJ, Doody DR, Biggs ML, Weiss NS, Starr JR, Schwartz SM. Physical activity in adolescence and testicular germ cell cancer risk. Cancer Causes Control. 2009;20(8):1281–90. https://doi.org/10.1007/s10552-009-9347-6 .

Coldman AJ, Elwood JM, Gallagher RP. Sports activities and risk of testicular cancer. Br J Cancer. 1982;46(5):749–56. https://doi.org/10.1038/bjc.1982.267 .

Hosseini M, SeyedAlinaghi S, Mahmoudi M, McFarland W. A case-control study of risk factors for prostate cancer in Iran. Acta Med Iran. 2010;48(1):61–6.

Pang Y, Lv J, Kartsonaki C, et al. Association of physical activity with risk of hepatobiliary diseases in China: a prospective cohort study of 0.5 million people. Br J Sports Med. 2021;55(18):1024–33. https://doi.org/10.1136/bjsports-2020-102174 .

Xiao Q, Liao L, Matthews CE, et al. Physical activity and renal cell carcinoma among black and white americans: a case-control study. BMC Cancer. 2014;14(1):707. https://doi.org/10.1186/1471-2407-14-707 .

Patterson R, Panter J, Vamos EP, Cummins S, Millett C, Laverty AA. Associations between commute mode and cardiovascular disease, cancer, and all-cause mortality, and cancer incidence, using linked Census data over 25 years in England and Wales: a cohort study. Lancet Planet Health. 2020;4(5):e186–94. https://doi.org/10.1016/S2542-5196(20)30079-6 .

Sahlqvist S, Goodman A, Simmons RK, et al. The association of cycling with all-cause, cardiovascular and cancer mortality: findings from the population-based EPIC-Norfolk cohort. BMJ Open. 2013;3(11):e003797. https://doi.org/10.1136/bmjopen-2013-003797 .

Autenrieth CS, Baumert J, Baumeister SE, et al. Association between domains of physical activity and all-cause, cardiovascular and cancer mortality. Eur J Epidemiol. 2011;26(2):91–9. https://doi.org/10.1007/s10654-010-9517-6 .

Batty GD, Shipley MJ, Marmot M, Smith GD. Physical activity and cause-specific mortality in men: further evidence from the Whitehall study. Eur J Epidemiol. 2001;17(9):863–9. https://doi.org/10.1023/A:1015609909969 .

Wu Y, Zhang D, Kang S. Physical activity and risk of breast cancer: a meta-analysis of prospective studies. Breast Cancer Res Treat. 2013;137(3):869–82. https://doi.org/10.1007/s10549-012-2396-7 .

Moore SC, Gierach GL, Schatzkin A, Matthews CE. Physical activity, sedentary behaviours, and the prevention of endometrial cancer. Br J Cancer. 2010;103(7):933–8. https://doi.org/10.1038/sj.bjc.6605902 .

García-Estévez L, Cortés J, Pérez S, Calvo I, Gallegos I, Moreno-Bueno G. Obesity and breast Cancer: a paradoxical and controversial relationship influenced by Menopausal Status. Front Oncol. 2021;11:705911. https://doi.org/10.3389/fonc.2021.705911 .

Chan DSM, Abar L, Cariolou M, et al. World Cancer Research Fund International: continuous update project—systematic literature review and meta-analysis of observational cohort studies on physical activity, sedentary behavior, adiposity, and weight change and breast cancer risk. Cancer Causes Control. 2019;30(11):1183–200. https://doi.org/10.1007/s10552-019-01223-w .

Compedium of Physical Activities. 17-Walking. Accessed August 3. 2023. https://sites.google.com/site/compendiumofphysicalactivities/Activity-Categories/walking?authuser=0 .

Mahmood S, MacInnis RJ, English DR, Karahalios A, Lynch BM. Domain-specific physical activity and sedentary behaviour in relation to colon and rectal cancer risk: a systematic review and meta-analysis. Int J Epidemiol. 2017;46(6):1797–813. https://doi.org/10.1093/ije/dyx137 .

Samad AKA, Taylor RS, Marshall T, Chapman MAS. A meta-analysis of the association of physical activity with reduced risk of colorectal cancer. Colorect Dis. 2005;7(3):204–13. https://doi.org/10.1111/j.1463-1318.2005.00747.x .

McTiernan A. Mechanisms linking physical activity with cancer. Nat Rev Cancer. 2008;8(3):205–11. https://doi.org/10.1038/nrc2325 .

Peters HPF. Potential benefits and hazards of physical activity and exercise on the gastrointestinal tract. Gut. 2001;48(3):435–9. https://doi.org/10.1136/gut.48.3.435 .

Diao X, Ling Y, Zeng Y, et al. Physical activity and cancer risk: a dose-response analysis for the global burden of Disease Study 2019. Cancer Commun. 2023;43(11):1229–43. https://doi.org/10.1002/cac2.12488 .

Garcia L, Pearce M, Abbas A, et al. Non-occupational physical activity and risk of cardiovascular disease, cancer and mortality outcomes: a dose–response meta-analysis of large prospective studies. Br J Sports Med. 2023;57(15):979–89. https://doi.org/10.1136/bjsports-2022-105669 .

Li T, Wei S, Shi Y, et al. The dose–response effect of physical activity on cancer mortality: findings from 71 prospective cohort studies. Br J Sports Med. 2016;50(6):339–45. https://doi.org/10.1136/bjsports-2015-094927 .

Unite, Nations. Department of Economic and Social Affair. World urbanization prospects: the 2018 revision. United Nations; 2019.

Sung H, Ferlay J, Siegel RL, Cancer, et al. J Clin. 2021;71(3):209–49. https://doi.org/10.3322/caac.21660 .

Download references

Acknowledgements

STT is supported by Sir Charles Hercus Health Research Fellowship from the Health Research Council of New Zealand (Ref: 23/051).

STT is supported by Sir Charles Hercus Health Research Fellowship from the Health Research Council of New Zealand (Ref: 23/051). The funder had no role in the study design, data collection, data analysis, data interpretation, writing of the report, approval of the manuscript, or decision to submit the manuscript for publication.

Author information

Authors and affiliations.

Epidemiology and Biostatistics, School of Population Health, University of Auckland, Auckland, New Zealand

Win Thu, Alistair Woodward, Alana Cavadino & Sandar Tin Tin

Cancer Epidemiology Unit, Oxford Population Health, University of Oxford, Oxford, UK

Sandar Tin Tin

You can also search for this author in PubMed   Google Scholar

Contributions

STT, AW, WT designed the study. STT supervised the study. WT conducted literature search, data analysis and wrote the original draft with critical inputs from STT, AC, and AW. All authors had full access to all the data in the study and had final responsibility for the decision to submit for publication. More than one author has directly accessed and verified the underlying data reported in the manuscript.

Corresponding author

Correspondence to Sandar Tin Tin .

Ethics declarations

Ethics approval and consent to participate.

Not applicable.

Consent for publication

Competing interests.

The authors declare no competing interests.

Additional information

Publisher’s note.

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Electronic supplementary material

Below is the link to the electronic supplementary material.

Supplementary Material 1

: Supplementary file S1 PRISMA checklist. Supplementary file S2 Literature search strategy. Supplementary file S3 List of excluded full texts with reasons. Supplementary file S4 Metabolic Equivalent of Task (MET) values used and MET hour per week conversion formulas. Supplementary file S5 Measurement units, effect measures and covariates included in the studies. Supplementary file S6 Newcastle-Ottawa Score of the studies (cohort studies). Supplementary file S7 Newcastle-Ottawa Score of the studies (case control studies). Supplementary file S8 Risks estimates used in the meta-analyses (separate excel sheet). Supplementary file S9 Forest plots. Supplementary file S10 Sub-group and covariates adjustment analyses. Supplementary file S11 Sensitivity analysis. Supplementary file S12 Funnel plots

Rights and permissions

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ . The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/ ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

Reprints and permissions

About this article

Cite this article.

Thu, W., Woodward, A., Cavadino, A. et al. Associations between transport modes and site-specific cancers: a systematic review and meta-analysis. Environ Health 23 , 39 (2024). https://doi.org/10.1186/s12940-024-01081-3

Download citation

Received : 20 November 2023

Accepted : 08 April 2024

Published : 13 April 2024

DOI : https://doi.org/10.1186/s12940-024-01081-3

Share this article

Anyone you share the following link with will be able to read this content:

Sorry, a shareable link is not currently available for this article.

Provided by the Springer Nature SharedIt content-sharing initiative

• Transport modes
• Active transport
• Site-specific cancers
• Systematic review
• Meta-analysis

Environmental Health

ISSN: 1476-069X

• General enquiries: [email protected]

An official website of the United States government

The .gov means it’s official. Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

The site is secure. The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

• Publications
• Account settings

Preview improvements coming to the PMC website in October 2024. Learn More or Try it out now .

• Advanced Search
• Journal List
• Lippincott Open Access
• PMC11005671

Clinical Case Study

Appendiceal mucinous neoplasms: a case report and review of literature.

Appendiceal mucinous neoplasms (AMNs) are uncommon tumors accounting for less than 1% of all cancers, which include a heterogeneous group of diseases with varying malignant potential. Pseudomyxoma peritonei (PMP) is a clinical syndrome characterized by progressive accumulation of mucinous tumor throughout the peritoneal cavity ( Carr et al., 2017 ). Because of difficulty in recognition during diagnosis, it is important to identify special clinical manifestations in AMNs patients. We here report a case of a patient diagnosed with AMN.

Case Presentation

A 60-year-old man was referred to our hospital because of ascites of unknown origin for 4 years. During these 4 years, he had abdominal distension and been diagnosed with liver cirrhosis. He had undergone relevant treatment, such as ascites drainage and diuretics, but without satisfactory effect. The patient had an otherwise insignificant previous medical history. On admission to our hospital, physical examination showed a fluid-filled abdomen; however, shifting dullness was not obvious. The abdomen was soft, with no direct tenderness or rebound tenderness.

The patient had stable vital signs. Blood and urine analyses were normal. Electrocardiography, chest radiography, and arterial blood gas analysis were also normal. Abdominal computed tomography (CT) suggested that there was a huge cystic lesion in the patient's abdomen and it seemed to be a lump containing solid and cystic components arising from the appendix (Figure ​ (Figure1). 1 ). The patient also underwent colonoscopy examination, which showed that the colon and rectum as well as orifice of vermiform appendix were normal (Figure ​ (Figure2 2 ).

Abdominal CT scan showing appendiceal lump (red arrow) and cystic lesion (yellow arrow) with intestinal tube extruded.

Colonoscopy showing normal orifice of vermiform appendix.

To identify the cause of the illness, subsequent exploratory laparotomy was performed. During the operation, a large amount of yellow and jelly-like turbid liquid (about 4,000 ml) was observed in the abdominal cavity (Figure ​ (Figure3). 3 ). After eliminating these abnormal substances, an ileocecal appendix lump (Figure ​ (Figure4) 4 ) appeared. The appendix lump was removed subsequently.

A large amount of yellow and jelly-like turbid liquid from the abdominal cavity.

The appendix neoplasm (5 cm × 4 cm × 3 cm) during surgery.

Microscopically, the pathology of the appendix lump tissue (Figure ​ (Figure5) 5 ) suggested hyperplasia of the mucous columnar epithelium and the mucus lake, which was formed by a large amount of abnormal mucus. The pathology of peritoneal nodules showed that there was a large amount of mucous tissue in the hyperplastic fibrous tissue. The final diagnosis was low-grade AMN (LAMN) accompanied with PMP.

(H&E, ×40 magnification) Histopathology of the appendix neoplasm showing hyperplasia of the mucous columnar epithelium (black arrow) and the mucus lake (white arrow).

After confirming the diagnosis, the patient underwent a cytoreductive surgery (CRS), and 1 month after operation, repeat CT scan showed no abnormalities (Figure ​ (Figure6). 6 ). Three months after CRS, the patient underwent hyperthermic intraperitoneal chemotherapy (HIPEC) with paclitaxel and cisplatin. Furthermore, we conducted telephone follow-up every 6 months until April of this year, and the patient was alive and in good condition.

The normal abdominal CT scan results after surgery.

AMNs are uncommon neoplasms that are detected in only 1% of appendectomy specimens ( Fournier et al., 2017 ). Because of the low-incidence rate and atypical clinical presentation, these patients may be misdiagnosed in clinical practice. Most cases are diagnosed incidentally at the time of appendicitis surgery or a screening CT. In this report, the patient was diagnosed as having ascites caused by cirrhosis for 4 years. However, the diagnosis of AMN was eventually confirmed by CT and surgery combined with pathology. Therefore, it is crucial to distinguish between ascites and pseudoascites (PA).

PA is an uncommon cause of abdominal distention mimicking ascites. When PA is caused by large abdominal cysts, it is difficult to distinguish from simple ascites because of the presence of an ultrathin wall and large liquid collection ( Rossi et al., 2019 ). Shifting dullness and a ruler-pressing test are typical clinical signs of ascites upon physical examination. The typical radiological signs of PA—the fact that the loops appear conglomerated in the center of the abdomen, or the fact that there may be aspects of septations in the fluid collection—are not a consistent finding at imaging and also they are not specific ( Camilon & Chilstrom, 2014 ; Nett, Vo, & Chapman, 2015 ; Shafi, Malla, & Reshi, 2009 ). Ascites from liver cirrhosis appears on the basis of the symptoms and signs of liver function damage. Although surgery combined with pathology examination is the criterion standard for diagnosis, it is beneficial for patients to undergo a differential procedure before an invasive surgery.

According to the new 2016 consensus statement from the Peritoneal Surface Oncology Group International (PSOGI), mucinous neoplasms are classified as follows ( Carr et al., 2016 ): LAMN, high-grade AMN (HAMN), mucinous adenocarcinoma, and poorly differentiated mucinous adenocarcinoma with signet ring features. LAMN extends beyond the mucosa into the appendiceal wall but without infiltrative invasion and with low-grade cytologic atypia ( Carr et al., 2016 ). Regardless of the degree of cellular atypia, both LAMN and HAMN can develop transmural extension or can rupture. When this occurs, the patient is at risk for developing PMP ( Bartlett et al., 2019 ).

This patient was finally diagnosed with LAMN accompanied with PMP by pathology examination. In recent years, a consensus both domestically and internationally recommends the integrated therapy of CRS and HIPEC as the standard treatment for PMP ( Li, Yu, & Liu, 2014 ; Yang et al., 2019 ). Therefore, this patient received HIPEC treatment 3 months after surgery, rather than immediately following CRS. In subsequent telephone follow-up every 6 months until April of this year, the patient did not experience any discomfort or recurrence. These results indicate that similar to the effect of immediate HIPEC after surgery, treatment with HIPEC 3 months after surgery may still achieve satisfactory results. Further large-scale, multicenter clinical studies are required to explore this point.

It is of great importance to identify ascites related to PA for patients diagnosed with AMN accompanied by PMP. Firmly mastering basic knowledge and skill of physical examination and radiographic imaging is a necessary quality for modern clinicians. In addition, early and accurate diagnosis will provide a clear direction for early treatment, thereby laying a solid foundation for improving the patient's prognosis.

THE OFFICIAL JOURNAL OF THE SOCIETY OF GASTROENTEROLOGY NURSES AND ASSOCIATES, INC.

DEDICATED TO THE SAFE AND EFFECTIVE PRACTICE OF GASTROENTEROLOGY AND ENDOSCOPY NURSING

The authors declare no conflicts of interest.

• Bartlett D. J., Thacker P. G., Grotz T. E., Graham R. P., Fletcher J. G., VanBuren W. M., Sheedy S. P. (2019). Mucinous appendiceal neoplasms: Classification, imaging, and HIPEC . Abdominal Radiology , 44 ( 5 ), 1686–1702. doi:10.1007/s00261-018-01888-y [ PubMed ] [ Google Scholar ]
• Camilon M., Chilstrom M. (2014). Ultrasound distinguishes ascites from a large ovarian fluid-filled cyst . The Western Journal of Emergency Medicine , 15 ( 7 ), 831. doi:10.5811/westjem.2014.9.23630 [ PMC free article ] [ PubMed ] [ Google Scholar ]
• Carr N. J., Bibeau F., Bradley R. F., Dartigues P., Feakins R. M., Geisinger K. R., Yantiss R. K. (2017). The histopathological classification, diagnosis and differential diagnosis of mucinous appendiceal neoplasms, appendiceal adenocarcinomas and pseudomyxoma peritonei . Histopathology , 71 ( 6 ), 847–858. doi:10.1111/his.13324 [ PubMed ] [ Google Scholar ]
• Carr N. J., Cecil T. D., Mohamed F., Sobin L. H., Sugarbaker P. H., González-Moreno S., Moran B. J.; Peritoneal Surface Oncology Group International. (2016). Peritoneal Surface Oncology Group I. A consensus for classification and pathologic reporting of pseudomyxoma peritonei and associated appendiceal neoplasia: The results of the Peritoneal Surface Oncology Group International (PSOGI) Modified Delphi Process . American Journal of Surgical Pathology , 40 ( 1 ), 14–26. doi:10.1097/PAS.0000000000000535 [ PubMed ] [ Google Scholar ]
• Fournier K., Rafeeq S., Taggart M., Kanaby P., Ning J., Chen H. C., Royal R. (2017). Low grade appendiceal mucinous neoplasm of uncertain malignant potential (LAMN-UMP): Prognostic factors and implications for treatment and follow-up . Annals of Surgical Oncology , 24 ( 1 ), 187–193. doi:10.1245/s10434-016-5588-2 [ PubMed ] [ Google Scholar ]
• Li Y., Yu Y., Liu Y. (2014). Report on the 9(th) International Congress on Peritoneal Surface Malignancies . Cancer Biology & Medicine , 11 ( 4 ), 281–284. doi:10.7497/j.issn.2095-3941.2014.04.008 [ PMC free article ] [ PubMed ] [ Google Scholar ]
• Nett M. H., Vo N. J., Chapman T. (2015). Large omental cyst . Radiology Case Reports , 5 ( 2 ), 388. doi:10.2484/rcr.v5i2.388 [ PMC free article ] [ PubMed ] [ Google Scholar ]
• Rossi A., Di Dato F., Iorio R., Vallone G., Mollica C., Caprio M. G., Spagnuolo M. I. (2019). Case report: Horse or zebra, ascites or pseudo-ascites? Care for pictural details ! BMC Pediatrics , 19 ( 1 ), 460. doi:10.1186/s12887-019-1826-7 [ PMC free article ] [ PubMed ] [ Google Scholar ]
• Shafi S. M., Malla M. A., Reshi F. A. (2009). Giant primary omental cyst mimicking a pseudoascites . African Journal of Paediatric Surgery , 6 ( 1 ), 58–60. doi:10.4103/0189-6725.48581 [ PubMed ] [ Google Scholar ]
• Yang L., Hongbin X., Zheng P., Shuzhong C., Wei W., Han L., Shaozhong W., et al. (2019). Expert consensus on cytoreductive surgery combined with hyperthermic intraperitoneal chemotherapy . National Medical Journal of China , 99 ( 20 ), 1527–1535. doi:10.3760/cma.j.issn.0376-2491.2019.20.003 [ Google Scholar ]