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What is a Literature Review? | Guide, Template, & Examples

Published on 22 February 2022 by Shona McCombes . Revised on 7 June 2022.

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.

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 summarise sources – it analyses, synthesises, and critically evaluates to give a clear picture of the state of knowledge on the subject.

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Table of contents

Why write 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, frequently asked questions about literature reviews, introduction.

• Quick Run-through
• Step 1 & 2

When you write a dissertation or thesis, you will 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 scholarly context
• Develop a theoretical framework and methodology for your research
• Position yourself in relation to other researchers and theorists
• Show how your dissertation addresses a gap or contributes to a debate

You might also have to write a literature review as a stand-alone assignment. In this case, the purpose is to evaluate the current state of research and demonstrate your knowledge of scholarly debates around a topic.

The content will look slightly different in each case, but the process of conducting a literature review follows the same steps. We’ve written a step-by-step guide that you can follow below.

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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 objectives and questions .

If you are writing a literature review as a stand-alone assignment, you will have to choose a focus and develop a central question to direct your search. Unlike a dissertation research question, this question has to be answerable without collecting original data. You should be able to answer it based only on a review of existing publications.

Make a list of keywords

Start by creating a list of keywords related to your research topic. 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 if 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 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 use boolean operators to help narrow down your search:

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.

To identify the most important publications on your topic, take note of recurring citations. If the same authors, books or articles keep appearing in your reading, make sure to seek them out.

You probably won’t be able to read absolutely everything that has been written on the topic – you’ll have to evaluate which sources are most relevant to your questions.

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?
• How does the publication contribute to your understanding of the topic? What are its key insights and arguments?
• 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 find out how many times an article has been cited on Google Scholar – a high citation count means the article has been influential in the field, and should certainly be included in your literature review.

The scope of your review will depend on your topic and discipline: in the sciences you usually only review recent literature, but in the humanities you might take a long historical perspective (for example, to trace how a concept has changed in meaning over time).

Remember that you can use our template to summarise and evaluate sources you’re thinking about using!

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’s important to keep track of your sources with references to avoid plagiarism . It can be helpful to make an annotated bibliography, where you compile full reference 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.

You can use our free APA Reference Generator for quick, correct, consistent citations.

Prevent plagiarism, run a free check.

To begin organising your literature review’s argument and structure, you need to 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 organising the body of a literature review. You should have a rough idea of your strategy before you start writing.

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 summarising sources in order.

Try to analyse 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 organise 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.

If you are writing the literature review as part of your dissertation or thesis, reiterate your central problem or research question and give a brief summary of the scholarly context. You can emphasise the timeliness of the topic (“many recent studies have focused on the problem of x”) or highlight a gap in the literature (“while there has been much research on x, few researchers have taken y into consideration”).

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, make sure to follow these tips:

• Summarise and synthesise: give an overview of the main points of each source and combine them into a coherent whole.
• Analyse and interpret: don’t just paraphrase other researchers – add your own interpretations, 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 transitions and topic sentences to draw connections, comparisons and contrasts.

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

If the literature review is part of your dissertation or thesis, reiterate how your research addresses gaps and contributes new knowledge, or discuss how you have drawn on existing theories and methods to build a framework for your research. This can lead directly into your methodology section.

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 dissertation , thesis, research paper , or proposal .

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

• To familiarise 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  dissertation . After the introduction , it grounds your research in a scholarly field and leads directly to your theoretical framework or methodology .

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• CAREER FEATURE
• 04 December 2020
• Correction 09 December 2020

How to write a superb literature review

Andy Tay is a freelance writer based in Singapore.

You can also search for this author in PubMed   Google Scholar

Literature reviews are important resources for scientists. They provide historical context for a field while offering opinions on its future trajectory. Creating them can provide inspiration for one’s own research, as well as some practice in writing. But few scientists are trained in how to write a review — or in what constitutes an excellent one. Even picking the appropriate software to use can be an involved decision (see ‘Tools and techniques’). So Nature asked editors and working scientists with well-cited reviews for their tips.

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doi: https://doi.org/10.1038/d41586-020-03422-x

Interviews have been edited for length and clarity.

Updates & Corrections

Correction 09 December 2020 : An earlier version of the tables in this article included some incorrect details about the programs Zotero, Endnote and Manubot. These have now been corrected.

Hsing, I.-M., Xu, Y. & Zhao, W. Electroanalysis 19 , 755–768 (2007).

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Ledesma, H. A. et al. Nature Nanotechnol. 14 , 645–657 (2019).

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Brahlek, M., Koirala, N., Bansal, N. & Oh, S. Solid State Commun. 215–216 , 54–62 (2015).

Choi, Y. & Lee, S. Y. Nature Rev. Chem . https://doi.org/10.1038/s41570-020-00221-w (2020).

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Ten Simple Rules for Writing a Literature Review

* E-mail: [email protected]

Affiliations Centre for Functional and Evolutionary Ecology (CEFE), CNRS, Montpellier, France, Centre for Biodiversity Synthesis and Analysis (CESAB), FRB, Aix-en-Provence, France

• Marco Pautasso

Published: July 18, 2013

• https://doi.org/10.1371/journal.pcbi.1003149
• Reader Comments

Citation: Pautasso M (2013) Ten Simple Rules for Writing a Literature Review. PLoS Comput Biol 9(7): e1003149. https://doi.org/10.1371/journal.pcbi.1003149

Editor: Philip E. Bourne, University of California San Diego, United States of America

Copyright: © 2013 Marco Pautasso. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Funding: This work was funded by the French Foundation for Research on Biodiversity (FRB) through its Centre for Synthesis and Analysis of Biodiversity data (CESAB), as part of the NETSEED research project. The funders had no role in the preparation of the manuscript.

Competing interests: The author has declared that no competing interests exist.

Literature reviews are in great demand in most scientific fields. Their need stems from the ever-increasing output of scientific publications [1] . For example, compared to 1991, in 2008 three, eight, and forty times more papers were indexed in Web of Science on malaria, obesity, and biodiversity, respectively [2] . Given such mountains of papers, scientists cannot be expected to examine in detail every single new paper relevant to their interests [3] . Thus, it is both advantageous and necessary to rely on regular summaries of the recent literature. Although recognition for scientists mainly comes from primary research, timely literature reviews can lead to new synthetic insights and are often widely read [4] . For such summaries to be useful, however, they need to be compiled in a professional way [5] .

When starting from scratch, reviewing the literature can require a titanic amount of work. That is why researchers who have spent their career working on a certain research issue are in a perfect position to review that literature. Some graduate schools are now offering courses in reviewing the literature, given that most research students start their project by producing an overview of what has already been done on their research issue [6] . However, it is likely that most scientists have not thought in detail about how to approach and carry out a literature review.

Reviewing the literature requires the ability to juggle multiple tasks, from finding and evaluating relevant material to synthesising information from various sources, from critical thinking to paraphrasing, evaluating, and citation skills [7] . In this contribution, I share ten simple rules I learned working on about 25 literature reviews as a PhD and postdoctoral student. Ideas and insights also come from discussions with coauthors and colleagues, as well as feedback from reviewers and editors.

Rule 1: Define a Topic and Audience

How to choose which topic to review? There are so many issues in contemporary science that you could spend a lifetime of attending conferences and reading the literature just pondering what to review. On the one hand, if you take several years to choose, several other people may have had the same idea in the meantime. On the other hand, only a well-considered topic is likely to lead to a brilliant literature review [8] . The topic must at least be:

• interesting to you (ideally, you should have come across a series of recent papers related to your line of work that call for a critical summary),
• an important aspect of the field (so that many readers will be interested in the review and there will be enough material to write it), and
• a well-defined issue (otherwise you could potentially include thousands of publications, which would make the review unhelpful).

Ideas for potential reviews may come from papers providing lists of key research questions to be answered [9] , but also from serendipitous moments during desultory reading and discussions. In addition to choosing your topic, you should also select a target audience. In many cases, the topic (e.g., web services in computational biology) will automatically define an audience (e.g., computational biologists), but that same topic may also be of interest to neighbouring fields (e.g., computer science, biology, etc.).

Rule 2: Search and Re-search the Literature

After having chosen your topic and audience, start by checking the literature and downloading relevant papers. Five pieces of advice here:

• keep track of the search items you use (so that your search can be replicated [10] ),
• keep a list of papers whose pdfs you cannot access immediately (so as to retrieve them later with alternative strategies),
• use a paper management system (e.g., Mendeley, Papers, Qiqqa, Sente),
• define early in the process some criteria for exclusion of irrelevant papers (these criteria can then be described in the review to help define its scope), and
• do not just look for research papers in the area you wish to review, but also seek previous reviews.

The chances are high that someone will already have published a literature review ( Figure 1 ), if not exactly on the issue you are planning to tackle, at least on a related topic. If there are already a few or several reviews of the literature on your issue, my advice is not to give up, but to carry on with your own literature review,

• PPT PowerPoint slide
• PNG larger image
• TIFF original image

The bottom-right situation (many literature reviews but few research papers) is not just a theoretical situation; it applies, for example, to the study of the impacts of climate change on plant diseases, where there appear to be more literature reviews than research studies [33] .

https://doi.org/10.1371/journal.pcbi.1003149.g001

• discussing in your review the approaches, limitations, and conclusions of past reviews,
• trying to find a new angle that has not been covered adequately in the previous reviews, and
• incorporating new material that has inevitably accumulated since their appearance.

When searching the literature for pertinent papers and reviews, the usual rules apply:

• be thorough,
• use different keywords and database sources (e.g., DBLP, Google Scholar, ISI Proceedings, JSTOR Search, Medline, Scopus, Web of Science), and
• look at who has cited past relevant papers and book chapters.

Rule 3: Take Notes While Reading

If you read the papers first, and only afterwards start writing the review, you will need a very good memory to remember who wrote what, and what your impressions and associations were while reading each single paper. My advice is, while reading, to start writing down interesting pieces of information, insights about how to organize the review, and thoughts on what to write. This way, by the time you have read the literature you selected, you will already have a rough draft of the review.

Of course, this draft will still need much rewriting, restructuring, and rethinking to obtain a text with a coherent argument [11] , but you will have avoided the danger posed by staring at a blank document. Be careful when taking notes to use quotation marks if you are provisionally copying verbatim from the literature. It is advisable then to reformulate such quotes with your own words in the final draft. It is important to be careful in noting the references already at this stage, so as to avoid misattributions. Using referencing software from the very beginning of your endeavour will save you time.

Rule 4: Choose the Type of Review You Wish to Write

After having taken notes while reading the literature, you will have a rough idea of the amount of material available for the review. This is probably a good time to decide whether to go for a mini- or a full review. Some journals are now favouring the publication of rather short reviews focusing on the last few years, with a limit on the number of words and citations. A mini-review is not necessarily a minor review: it may well attract more attention from busy readers, although it will inevitably simplify some issues and leave out some relevant material due to space limitations. A full review will have the advantage of more freedom to cover in detail the complexities of a particular scientific development, but may then be left in the pile of the very important papers “to be read” by readers with little time to spare for major monographs.

There is probably a continuum between mini- and full reviews. The same point applies to the dichotomy of descriptive vs. integrative reviews. While descriptive reviews focus on the methodology, findings, and interpretation of each reviewed study, integrative reviews attempt to find common ideas and concepts from the reviewed material [12] . A similar distinction exists between narrative and systematic reviews: while narrative reviews are qualitative, systematic reviews attempt to test a hypothesis based on the published evidence, which is gathered using a predefined protocol to reduce bias [13] , [14] . When systematic reviews analyse quantitative results in a quantitative way, they become meta-analyses. The choice between different review types will have to be made on a case-by-case basis, depending not just on the nature of the material found and the preferences of the target journal(s), but also on the time available to write the review and the number of coauthors [15] .

Rule 5: Keep the Review Focused, but Make It of Broad Interest

Whether your plan is to write a mini- or a full review, it is good advice to keep it focused 16 , 17 . Including material just for the sake of it can easily lead to reviews that are trying to do too many things at once. The need to keep a review focused can be problematic for interdisciplinary reviews, where the aim is to bridge the gap between fields [18] . If you are writing a review on, for example, how epidemiological approaches are used in modelling the spread of ideas, you may be inclined to include material from both parent fields, epidemiology and the study of cultural diffusion. This may be necessary to some extent, but in this case a focused review would only deal in detail with those studies at the interface between epidemiology and the spread of ideas.

While focus is an important feature of a successful review, this requirement has to be balanced with the need to make the review relevant to a broad audience. This square may be circled by discussing the wider implications of the reviewed topic for other disciplines.

Rule 6: Be Critical and Consistent

Reviewing the literature is not stamp collecting. A good review does not just summarize the literature, but discusses it critically, identifies methodological problems, and points out research gaps [19] . After having read a review of the literature, a reader should have a rough idea of:

• the major achievements in the reviewed field,
• the main areas of debate, and
• the outstanding research questions.

It is challenging to achieve a successful review on all these fronts. A solution can be to involve a set of complementary coauthors: some people are excellent at mapping what has been achieved, some others are very good at identifying dark clouds on the horizon, and some have instead a knack at predicting where solutions are going to come from. If your journal club has exactly this sort of team, then you should definitely write a review of the literature! In addition to critical thinking, a literature review needs consistency, for example in the choice of passive vs. active voice and present vs. past tense.

Rule 7: Find a Logical Structure

Like a well-baked cake, a good review has a number of telling features: it is worth the reader's time, timely, systematic, well written, focused, and critical. It also needs a good structure. With reviews, the usual subdivision of research papers into introduction, methods, results, and discussion does not work or is rarely used. However, a general introduction of the context and, toward the end, a recapitulation of the main points covered and take-home messages make sense also in the case of reviews. For systematic reviews, there is a trend towards including information about how the literature was searched (database, keywords, time limits) [20] .

How can you organize the flow of the main body of the review so that the reader will be drawn into and guided through it? It is generally helpful to draw a conceptual scheme of the review, e.g., with mind-mapping techniques. Such diagrams can help recognize a logical way to order and link the various sections of a review [21] . This is the case not just at the writing stage, but also for readers if the diagram is included in the review as a figure. A careful selection of diagrams and figures relevant to the reviewed topic can be very helpful to structure the text too [22] .

Rule 8: Make Use of Feedback

Reviews of the literature are normally peer-reviewed in the same way as research papers, and rightly so [23] . As a rule, incorporating feedback from reviewers greatly helps improve a review draft. Having read the review with a fresh mind, reviewers may spot inaccuracies, inconsistencies, and ambiguities that had not been noticed by the writers due to rereading the typescript too many times. It is however advisable to reread the draft one more time before submission, as a last-minute correction of typos, leaps, and muddled sentences may enable the reviewers to focus on providing advice on the content rather than the form.

Feedback is vital to writing a good review, and should be sought from a variety of colleagues, so as to obtain a diversity of views on the draft. This may lead in some cases to conflicting views on the merits of the paper, and on how to improve it, but such a situation is better than the absence of feedback. A diversity of feedback perspectives on a literature review can help identify where the consensus view stands in the landscape of the current scientific understanding of an issue [24] .

Rule 9: Include Your Own Relevant Research, but Be Objective

In many cases, reviewers of the literature will have published studies relevant to the review they are writing. This could create a conflict of interest: how can reviewers report objectively on their own work [25] ? Some scientists may be overly enthusiastic about what they have published, and thus risk giving too much importance to their own findings in the review. However, bias could also occur in the other direction: some scientists may be unduly dismissive of their own achievements, so that they will tend to downplay their contribution (if any) to a field when reviewing it.

In general, a review of the literature should neither be a public relations brochure nor an exercise in competitive self-denial. If a reviewer is up to the job of producing a well-organized and methodical review, which flows well and provides a service to the readership, then it should be possible to be objective in reviewing one's own relevant findings. In reviews written by multiple authors, this may be achieved by assigning the review of the results of a coauthor to different coauthors.

Rule 10: Be Up-to-Date, but Do Not Forget Older Studies

Given the progressive acceleration in the publication of scientific papers, today's reviews of the literature need awareness not just of the overall direction and achievements of a field of inquiry, but also of the latest studies, so as not to become out-of-date before they have been published. Ideally, a literature review should not identify as a major research gap an issue that has just been addressed in a series of papers in press (the same applies, of course, to older, overlooked studies (“sleeping beauties” [26] )). This implies that literature reviewers would do well to keep an eye on electronic lists of papers in press, given that it can take months before these appear in scientific databases. Some reviews declare that they have scanned the literature up to a certain point in time, but given that peer review can be a rather lengthy process, a full search for newly appeared literature at the revision stage may be worthwhile. Assessing the contribution of papers that have just appeared is particularly challenging, because there is little perspective with which to gauge their significance and impact on further research and society.

Inevitably, new papers on the reviewed topic (including independently written literature reviews) will appear from all quarters after the review has been published, so that there may soon be the need for an updated review. But this is the nature of science [27] – [32] . I wish everybody good luck with writing a review of the literature.

Acknowledgments

Many thanks to M. Barbosa, K. Dehnen-Schmutz, T. Döring, D. Fontaneto, M. Garbelotto, O. Holdenrieder, M. Jeger, D. Lonsdale, A. MacLeod, P. Mills, M. Moslonka-Lefebvre, G. Stancanelli, P. Weisberg, and X. Xu for insights and discussions, and to P. Bourne, T. Matoni, and D. Smith for helpful comments on a previous draft.

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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.

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What is a Literature Review? How to Write It (with Examples)

A literature review is a critical analysis and synthesis of existing research on a particular topic. It provides an overview of the current state of knowledge, identifies gaps, and highlights key findings in the literature. 1 The purpose of a literature review is to situate your own research within the context of existing scholarship, demonstrating your understanding of the topic and showing how your work contributes to the ongoing conversation in the field. Learning how to write a literature review is a critical tool for successful research. Your ability to summarize and synthesize prior research pertaining to a certain topic demonstrates your grasp on the topic of study, and assists in the learning process. 

Table of Contents

• What is the purpose of literature review? 
• a. Habitat Loss and Species Extinction: 
• b. Range Shifts and Phenological Changes: 
• c. Ocean Acidification and Coral Reefs: 
• d. Adaptive Strategies and Conservation Efforts: 
• How to write a good literature review 
• Choose a Topic and Define the Research Question: 
• Decide on the Scope of Your Review: 
• Select Databases for Searches: 
• Conduct Searches and Keep Track: 
• Review the Literature: 
• Organize and Write Your Literature Review: 
• Frequently asked questions 

What is a literature review?

A well-conducted literature review demonstrates the researcher’s familiarity with the existing literature, establishes the context for their own research, and contributes to scholarly conversations on the topic. One of the purposes of a literature review is also to help researchers avoid duplicating previous work and ensure that their research is informed by and builds upon the existing body of knowledge.

What is the purpose of literature review?

A literature review serves several important purposes within academic and research contexts. Here are some key objectives and functions of a literature review: 2  

• Contextualizing the Research Problem: The literature review provides a background and context for the research problem under investigation. It helps to situate the study within the existing body of knowledge. 
• Identifying Gaps in Knowledge: By identifying gaps, contradictions, or areas requiring further research, the researcher can shape the research question and justify the significance of the study. This is crucial for ensuring that the new research contributes something novel to the field. 
• Understanding Theoretical and Conceptual Frameworks: Literature reviews help researchers gain an understanding of the theoretical and conceptual frameworks used in previous studies. This aids in the development of a theoretical framework for the current research. 
• Providing Methodological Insights: Another purpose of literature reviews is that it allows researchers to learn about the methodologies employed in previous studies. This can help in choosing appropriate research methods for the current study and avoiding pitfalls that others may have encountered. 
• Establishing Credibility: A well-conducted literature review demonstrates the researcher’s familiarity with existing scholarship, establishing their credibility and expertise in the field. It also helps in building a solid foundation for the new research. 
• Informing Hypotheses or Research Questions: The literature review guides the formulation of hypotheses or research questions by highlighting relevant findings and areas of uncertainty in existing literature. 

Literature review example

Let’s delve deeper with a literature review example: Let’s say your literature review is about the impact of climate change on biodiversity. You might format your literature review into sections such as the effects of climate change on habitat loss and species extinction, phenological changes, and marine biodiversity. Each section would then summarize and analyze relevant studies in those areas, highlighting key findings and identifying gaps in the research. The review would conclude by emphasizing the need for further research on specific aspects of the relationship between climate change and biodiversity. The following literature review template provides a glimpse into the recommended literature review structure and content, demonstrating how research findings are organized around specific themes within a broader topic. 

Literature Review on Climate Change Impacts on Biodiversity:

Climate change is a global phenomenon with far-reaching consequences, including significant impacts on biodiversity. This literature review synthesizes key findings from various studies: 

a. Habitat Loss and Species Extinction:

Climate change-induced alterations in temperature and precipitation patterns contribute to habitat loss, affecting numerous species (Thomas et al., 2004). The review discusses how these changes increase the risk of extinction, particularly for species with specific habitat requirements. 

b. Range Shifts and Phenological Changes:

Observations of range shifts and changes in the timing of biological events (phenology) are documented in response to changing climatic conditions (Parmesan & Yohe, 2003). These shifts affect ecosystems and may lead to mismatches between species and their resources. 

c. Ocean Acidification and Coral Reefs:

The review explores the impact of climate change on marine biodiversity, emphasizing ocean acidification’s threat to coral reefs (Hoegh-Guldberg et al., 2007). Changes in pH levels negatively affect coral calcification, disrupting the delicate balance of marine ecosystems. 

d. Adaptive Strategies and Conservation Efforts:

Recognizing the urgency of the situation, the literature review discusses various adaptive strategies adopted by species and conservation efforts aimed at mitigating the impacts of climate change on biodiversity (Hannah et al., 2007). It emphasizes the importance of interdisciplinary approaches for effective conservation planning. 

How to write a good literature review

Writing a literature review involves summarizing and synthesizing existing research on a particular topic. A good literature review format should include the following elements. 

Introduction: The introduction sets the stage for your literature review, providing context and introducing the main focus of your review. 

• Opening Statement: Begin with a general statement about the broader topic and its significance in the field. 
• Scope and Purpose: Clearly define the scope of your literature review. Explain the specific research question or objective you aim to address. 
• Organizational Framework: Briefly outline the structure of your literature review, indicating how you will categorize and discuss the existing research. 
• Significance of the Study: Highlight why your literature review is important and how it contributes to the understanding of the chosen topic. 
• Thesis Statement: Conclude the introduction with a concise thesis statement that outlines the main argument or perspective you will develop in the body of the literature review. 

Body: The body of the literature review is where you provide a comprehensive analysis of existing literature, grouping studies based on themes, methodologies, or other relevant criteria. 

• Organize by Theme or Concept: Group studies that share common themes, concepts, or methodologies. Discuss each theme or concept in detail, summarizing key findings and identifying gaps or areas of disagreement. 
• Critical Analysis: Evaluate the strengths and weaknesses of each study. Discuss the methodologies used, the quality of evidence, and the overall contribution of each work to the understanding of the topic. 
• Synthesis of Findings: Synthesize the information from different studies to highlight trends, patterns, or areas of consensus in the literature. 
• Identification of Gaps: Discuss any gaps or limitations in the existing research and explain how your review contributes to filling these gaps. 
• Transition between Sections: Provide smooth transitions between different themes or concepts to maintain the flow of your literature review. 

Conclusion: The conclusion of your literature review should summarize the main findings, highlight the contributions of the review, and suggest avenues for future research. 

• Summary of Key Findings: Recap the main findings from the literature and restate how they contribute to your research question or objective. 
• Contributions to the Field: Discuss the overall contribution of your literature review to the existing knowledge in the field. 
• Implications and Applications: Explore the practical implications of the findings and suggest how they might impact future research or practice. 
• Recommendations for Future Research: Identify areas that require further investigation and propose potential directions for future research in the field. 
• Final Thoughts: Conclude with a final reflection on the importance of your literature review and its relevance to the broader academic community. 

Conducting a literature review

Conducting a literature review is an essential step in research that involves reviewing and analyzing existing literature on a specific topic. It’s important to know how to do a literature review effectively, so here are the steps to follow: 1  

Choose a Topic and Define the Research Question:

• Select a topic that is relevant to your field of study. 
• Clearly define your research question or objective. Determine what specific aspect of the topic do you want to explore? 

Decide on the Scope of Your Review:

• Determine the timeframe for your literature review. Are you focusing on recent developments, or do you want a historical overview? 
• Consider the geographical scope. Is your review global, or are you focusing on a specific region? 
• Define the inclusion and exclusion criteria. What types of sources will you include? Are there specific types of studies or publications you will exclude? 

Select Databases for Searches:

• Identify relevant databases for your field. Examples include PubMed, IEEE Xplore, Scopus, Web of Science, and Google Scholar. 
• Consider searching in library catalogs, institutional repositories, and specialized databases related to your topic. 

Conduct Searches and Keep Track:

• Develop a systematic search strategy using keywords, Boolean operators (AND, OR, NOT), and other search techniques. 
• Record and document your search strategy for transparency and replicability. 
• Keep track of the articles, including publication details, abstracts, and links. Use citation management tools like EndNote, Zotero, or Mendeley to organize your references. 

Review the Literature:

• Evaluate the relevance and quality of each source. Consider the methodology, sample size, and results of studies. 
• Organize the literature by themes or key concepts. Identify patterns, trends, and gaps in the existing research. 
• Summarize key findings and arguments from each source. Compare and contrast different perspectives. 
• Identify areas where there is a consensus in the literature and where there are conflicting opinions. 
• Provide critical analysis and synthesis of the literature. What are the strengths and weaknesses of existing research? 

Organize and Write Your Literature Review:

• Literature review outline should be based on themes, chronological order, or methodological approaches. 
• Write a clear and coherent narrative that synthesizes the information gathered. 
• Use proper citations for each source and ensure consistency in your citation style (APA, MLA, Chicago, etc.). 
• Conclude your literature review by summarizing key findings, identifying gaps, and suggesting areas for future research. 

The literature review sample and detailed advice on writing and conducting a review will help you produce a well-structured report. But remember that a literature review is an ongoing process, and it may be necessary to revisit and update it as your research progresses. 

Frequently asked questions

A literature review is a critical and comprehensive analysis of existing literature (published and unpublished works) on a specific topic or research question and provides a synthesis of the current state of knowledge in a particular field. A well-conducted literature review is crucial for researchers to build upon existing knowledge, avoid duplication of efforts, and contribute to the advancement of their field. It also helps researchers situate their work within a broader context and facilitates the development of a sound theoretical and conceptual framework for their studies.

Literature review is a crucial component of research writing, providing a solid background for a research paper’s investigation. The aim is to keep professionals up to date by providing an understanding of ongoing developments within a specific field, including research methods, and experimental techniques used in that field, and present that knowledge in the form of a written report. Also, the depth and breadth of the literature review emphasizes the credibility of the scholar in his or her field.  

Before writing a literature review, it’s essential to undertake several preparatory steps to ensure that your review is well-researched, organized, and focused. This includes choosing a topic of general interest to you and doing exploratory research on that topic, writing an annotated bibliography, and noting major points, especially those that relate to the position you have taken on the topic. 

Literature reviews and academic research papers are essential components of scholarly work but serve different purposes within the academic realm. 3 A literature review aims to provide a foundation for understanding the current state of research on a particular topic, identify gaps or controversies, and lay the groundwork for future research. Therefore, it draws heavily from existing academic sources, including books, journal articles, and other scholarly publications. In contrast, an academic research paper aims to present new knowledge, contribute to the academic discourse, and advance the understanding of a specific research question. Therefore, it involves a mix of existing literature (in the introduction and literature review sections) and original data or findings obtained through research methods. 

Literature reviews are essential components of academic and research papers, and various strategies can be employed to conduct them effectively. If you want to know how to write a literature review for a research paper, here are four common approaches that are often used by researchers.  Chronological Review: This strategy involves organizing the literature based on the chronological order of publication. It helps to trace the development of a topic over time, showing how ideas, theories, and research have evolved.  Thematic Review: Thematic reviews focus on identifying and analyzing themes or topics that cut across different studies. Instead of organizing the literature chronologically, it is grouped by key themes or concepts, allowing for a comprehensive exploration of various aspects of the topic.  Methodological Review: This strategy involves organizing the literature based on the research methods employed in different studies. It helps to highlight the strengths and weaknesses of various methodologies and allows the reader to evaluate the reliability and validity of the research findings.  Theoretical Review: A theoretical review examines the literature based on the theoretical frameworks used in different studies. This approach helps to identify the key theories that have been applied to the topic and assess their contributions to the understanding of the subject.  It’s important to note that these strategies are not mutually exclusive, and a literature review may combine elements of more than one approach. The choice of strategy depends on the research question, the nature of the literature available, and the goals of the review. Additionally, other strategies, such as integrative reviews or systematic reviews, may be employed depending on the specific requirements of the research.

The literature review format can vary depending on the specific publication guidelines. However, there are some common elements and structures that are often followed. Here is a general guideline for the format of a literature review:  Introduction:   Provide an overview of the topic.  Define the scope and purpose of the literature review.  State the research question or objective.  Body:   Organize the literature by themes, concepts, or chronology.  Critically analyze and evaluate each source.  Discuss the strengths and weaknesses of the studies.  Highlight any methodological limitations or biases.  Identify patterns, connections, or contradictions in the existing research.  Conclusion:   Summarize the key points discussed in the literature review.  Highlight the research gap.  Address the research question or objective stated in the introduction.  Highlight the contributions of the review and suggest directions for future research.

Both annotated bibliographies and literature reviews involve the examination of scholarly sources. While annotated bibliographies focus on individual sources with brief annotations, literature reviews provide a more in-depth, integrated, and comprehensive analysis of existing literature on a specific topic. The key differences are as follows: 

References 

• Denney, A. S., & Tewksbury, R. (2013). How to write a literature review.  Journal of criminal justice education ,  24 (2), 218-234. 
• Pan, M. L. (2016).  Preparing literature reviews: Qualitative and quantitative approaches . Taylor & Francis. 
• Cantero, C. (2019). How to write a literature review.  San José State University Writing Center . 

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Libraries | Research Guides

Literature reviews, what is a literature review, learning more about how to do a literature review.

• Planning the Review
• The Research Question
• Choosing Where to Search
• Organizing the Review
• Writing the Review

A literature review is a review and synthesis of existing research on a topic or research question. A literature review is meant to analyze the scholarly literature, make connections across writings and identify strengths, weaknesses, trends, and missing conversations. A literature review should address different aspects of a topic as it relates to your research question. A literature review goes beyond a description or summary of the literature you have read. 

• Sage Research Methods Core Collection This link opens in a new window SAGE Research Methods supports research at all levels by providing material to guide users through every step of the research process. SAGE Research Methods is the ultimate methods library with more than 1000 books, reference works, journal articles, and instructional videos by world-leading academics from across the social sciences, including the largest collection of qualitative methods books available online from any scholarly publisher. – Publisher

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How to Write a Literature Review

What is a literature review.

• What Is the Literature
• Writing the Review

A literature review is much more than an annotated bibliography or a list of separate reviews of articles and books. It is a critical, analytical summary and synthesis of the current knowledge of a topic. Thus it should compare and relate different theories, findings, etc, rather than just summarize them individually. In addition, it should have a particular focus or theme to organize the review. It does not have to be an exhaustive account of everything published on the topic, but it should discuss all the significant academic literature and other relevant sources important for that focus.

This is meant to be a general guide to writing a literature review: ways to structure one, what to include, how it supplements other research. For more specific help on writing a review, and especially for help on finding the literature to review, sign up for a Personal Research Session .

The specific organization of a literature review depends on the type and purpose of the review, as well as on the specific field or topic being reviewed. But in general, it is a relatively brief but thorough exploration of past and current work on a topic. Rather than a chronological listing of previous work, though, literature reviews are usually organized thematically, such as different theoretical approaches, methodologies, or specific issues or concepts involved in the topic. A thematic organization makes it much easier to examine contrasting perspectives, theoretical approaches, methodologies, findings, etc, and to analyze the strengths and weaknesses of, and point out any gaps in, previous research. And this is the heart of what a literature review is about. A literature review may offer new interpretations, theoretical approaches, or other ideas; if it is part of a research proposal or report it should demonstrate the relationship of the proposed or reported research to others' work; but whatever else it does, it must provide a critical overview of the current state of research efforts. 

Literature reviews are common and very important in the sciences and social sciences. They are less common and have a less important role in the humanities, but they do have a place, especially stand-alone reviews.

Types of Literature Reviews

There are different types of literature reviews, and different purposes for writing a review, but the most common are:

• Stand-alone literature review articles . These provide an overview and analysis of the current state of research on a topic or question. The goal is to evaluate and compare previous research on a topic to provide an analysis of what is currently known, and also to reveal controversies, weaknesses, and gaps in current work, thus pointing to directions for future research. You can find examples published in any number of academic journals, but there is a series of Annual Reviews of *Subject* which are specifically devoted to literature review articles. Writing a stand-alone review is often an effective way to get a good handle on a topic and to develop ideas for your own research program. For example, contrasting theoretical approaches or conflicting interpretations of findings can be the basis of your research project: can you find evidence supporting one interpretation against another, or can you propose an alternative interpretation that overcomes their limitations?
• Part of a research proposal . This could be a proposal for a PhD dissertation, a senior thesis, or a class project. It could also be a submission for a grant. The literature review, by pointing out the current issues and questions concerning a topic, is a crucial part of demonstrating how your proposed research will contribute to the field, and thus of convincing your thesis committee to allow you to pursue the topic of your interest or a funding agency to pay for your research efforts.
• Part of a research report . When you finish your research and write your thesis or paper to present your findings, it should include a literature review to provide the context to which your work is a contribution. Your report, in addition to detailing the methods, results, etc. of your research, should show how your work relates to others' work.

A literature review for a research report is often a revision of the review for a research proposal, which can be a revision of a stand-alone review. Each revision should be a fairly extensive revision. With the increased knowledge of and experience in the topic as you proceed, your understanding of the topic will increase. Thus, you will be in a better position to analyze and critique the literature. In addition, your focus will change as you proceed in your research. Some areas of the literature you initially reviewed will be marginal or irrelevant for your eventual research, and you will need to explore other areas more thoroughly. 

Examples of Literature Reviews

See the series of Annual Reviews of *Subject* which are specifically devoted to literature review articles to find many examples of stand-alone literature reviews in the biomedical, physical, and social sciences. 

Research report articles vary in how they are organized, but a common general structure is to have sections such as:

• Abstract - Brief summary of the contents of the article
• Introduction - A explanation of the purpose of the study, a statement of the research question(s) the study intends to address
• Literature review - A critical assessment of the work done so far on this topic, to show how the current study relates to what has already been done
• Methods - How the study was carried out (e.g. instruments or equipment, procedures, methods to gather and analyze data)
• Results - What was found in the course of the study
• Discussion - What do the results mean
• Conclusion - State the conclusions and implications of the results, and discuss how it relates to the work reviewed in the literature review; also, point to directions for further work in the area

Here are some articles that illustrate variations on this theme. There is no need to read the entire articles (unless the contents interest you); just quickly browse through to see the sections, and see how each section is introduced and what is contained in them.

The Determinants of Undergraduate Grade Point Average: The Relative Importance of Family Background, High School Resources, and Peer Group Effects , in The Journal of Human Resources , v. 34 no. 2 (Spring 1999), p. 268-293.

This article has a standard breakdown of sections:

• Introduction
• Literature Review
• Some discussion sections

First Encounters of the Bureaucratic Kind: Early Freshman Experiences with a Campus Bureaucracy , in The Journal of Higher Education , v. 67 no. 6 (Nov-Dec 1996), p. 660-691.

This one does not have a section specifically labeled as a "literature review" or "review of the literature," but the first few sections cite a long list of other sources discussing previous research in the area before the authors present their own study they are reporting.

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Scholarly Articles: How can I tell?

• Journal Information

Literature Review

• Author and affiliation
• Introduction
• Specialized Vocabulary
• Methodology
• Research sponsors
• Peer-review

The literature review section of an article is a summary or analysis of all the research the author read before doing his/her own research. This section may be part of the introduction or in a section called Background. It provides the background on who has done related research, what that research has or has not uncovered and how the current research contributes to the conversation on the topic. When you read the lit review ask:

• Does the review of the literature logically lead up to the research questions?
• Do the authors review articles relevant to their research study?
• Do the authors show where there are gaps in the literature?

The lit review is also a good place to find other sources you may want to read on this topic to help you get the bigger picture.

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How to Write a Literature Review

• Reading Journal Articles
• Literature Reviews: A Recap

How to Read and Comprehend Scientific Journal Articles

• Does it Describe a Literature Review?
• 1. Identify the Question
• 2. Review Discipline Styles
• Searching Article Databases
• Finding Full-Text of an Article
• Citation Chaining
• When to Stop Searching
• 4. Manage Your References
• 5. Critically Analyze and Evaluate
• 6. Synthesize
• 7. Write a Literature Review

Peer-reviewed or scholarly journal articles are written in a very specific organized and formal way. If you haven't spent much time with this type of literature, this video provides some tips for reading journal articles.

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• Published: 25 April 2024

A scoping review of academic and grey literature on migrant health research conducted in Scotland

• G. Petrie 1 ,
• K. Angus 2 &
• R. O’Donnell 2  

BMC Public Health volume  24 , Article number:  1156 ( 2024 ) Cite this article

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Migration to Scotland has increased since 2002 with an increase in European residents and participation in the Asylum dispersal scheme. Scotland has become more ethnically diverse, and 10% of the current population were born abroad. Migration and ethnicity are determinants of health, and information on the health status of migrants to Scotland and their access to and barriers to care facilitates the planning and delivery of equitable health services. This study aimed to scope existing peer-reviewed research and grey literature to identify gaps in evidence regarding the health of migrants in Scotland.

A scoping review on the health of migrants in Scotland was carried out for dates January 2002 to March 2023, inclusive of peer-reviewed journals and grey literature. CINAHL/ Web of Science/SocIndex and Medline databases were systematically searched along with government and third-sector websites. The searches identified 2166 journal articles and 170 grey literature documents for screening. Included articles were categorised according to the World Health Organisation’s 2016 Strategy and Action Plan for Refugee and Migrant Health in the European region. This approach builds on a previously published literature review on Migrant Health in the Republic of Ireland.

Seventy-one peer reviewed journal articles and 29 grey literature documents were included in the review. 66% were carried out from 2013 onwards and the majority focused on asylum seekers or unspecified migrant groups. Most research identified was on the World Health Organisation’s strategic areas of right to health of refugees, social determinants of health and public health planning and strengthening health systems. There were fewer studies on the strategic areas of frameworks for collaborative action, preventing communicable disease, preventing non-communicable disease, health screening and assessment and improving health information and communication.

While research on migrant health in Scotland has increased in recent years significant gaps remain. Future priorities should include studies of undocumented migrants, migrant workers, and additional research is required on the issue of improving health information and communication.

Peer Review reports

The term migrant is defined by the International Organisation for Migration as “ a person who moves away from his or her place of usual residence, whether within a country or across an international border, temporarily or permanently, and for a variety of reasons. The term includes several well-defined legal categories of people, including migrant workers; persons whose particular types of movements are legally-defined, such as smuggled migrants; as well as those whose status are not specifically defined under international law, such as international students.” [ 1 ] Internationally there are an estimated 281 million migrants – 3.6% of the world population, including 26.4 million refugees and 4.1 million asylum seekers – the highest number ever recorded [ 2 ]. The UN Refugee Society defines the term refugee as “ someone who has been forced to flee his or her country because of persecution, war or violence…most likely, they cannot return home or are afraid to do so .” The term asylum-seeker is defined as “someone whose request for sanctuary has yet to be processed.” [ 3 ].

Net-migration to Europe was negative in the 19th century due to higher levels of emigration, however in the mid-20th century immigration began to rise, because of an increase in migrant workers and following conflicts in the Middle East and North Africa [ 4 ]. Current migration drivers include conflicts alongside world-wide economic instability, exacerbated by the Covid-19 pandemic [ 5 ]. Environmental damage due to climate change is expected to inflate the number of asylum seekers entering Europe in future [ 6 ]. The increase in migration to Europe is not a short-term influx but a long-term phenomenon, and European nations must adapt and find solutions to resulting financial, safeguarding and health challenges [ 7 ].

Data on healthcare use by migrants in Europe is variable, which means cross-country comparisons are inadequate [ 8 ]. Many countries do not record migration information within health records and all use disparate criteria to classify migrant status. The lack of comparative data hinders public health surveillance and effective interventions [ 9 ]. Even where information is available, results can be contradictory due to the multifarious migrant population. Migrants have a wide range of origin countries, socio-economic position, age and journeys undertaken which can affect health status [ 10 ].

Migrants initially may have better health than the general population, known as the ‘Healthy Migrant effect’ [ 11 ]. However, health declines with increasing length of residence [ 12 ] and over time to levels comparable with the general population [ 13 ]. Second generation immigrants may have higher mortality than average [ 14 ]. The process of acculturation to the host country, with adoption of unhealthy lifestyle and behaviours, increases the risk for chronic disease [ 15 ]. In addition, inequalities in health of migrants compared to host populations has been confirmed by wide-ranging research [ 16 ].

Host countries may limit healthcare access, with undocumented migrants sometimes only entitled to emergency care [ 17 ]. Even when access is granted, inequitable services can affect quality of care due to language barriers and cultural factors [ 18 ]. Poor working/living conditions and discrimination can exacerbate health inequalities [ 12 ]. Processing facilities for asylum seekers are frequently overpopulated, stressful environments [ 19 ] and threat of deportation, lack of citizenship rights and integration can negatively affect health and access to care [ 20 ]. Undocumented workers are unprotected by health and safety legislation leading to dangerous working conditions and injuries [ 15 ].

A systematic review of migrant health in the European Union (EU) found migrants have worse self-perceived health than the general population [ 21 ]. Research evidence indicates increased prevalence of cardiovascular disease, diabetes, mental health disorders and adverse pregnancy outcomes. Exposure to conflict, harsh travel conditions and suboptimal vaccine programmes can mean higher risk of communicable disease [ 22 ]. Scoping reviews have also been conducted to describe trends within migration health research in the United Kingdom (UK) [ 23 ] and identify gaps for future research agendas in the UK [ 23 ] and in the Republic of Ireland [ 24 ].

Almost three-quarters (73%) of published migration health research in the UK has been conducted in England, focusing primarily on infectious diseases and mental health. There is limited evidence on the social determinants of health, access to and use of healthcare and structural and behavioural factors behaviours that influence migrant health in the UK [ 23 ]. By contrast, a large amount of the migration research conducted in the Republic of Ireland has focused on the social determinants of health, and on health system adaptations, with a paucity of research focusing on improving health information systems [ 24 ].

Migration and Health in Scotland

Immigration to Scotland began to rise in 2003 with the expansion of the EU [ 25 ]. The population in Scotland increased from 5.11 million to 5.47 million between 2005 and 2020 and is predicted to continue rising until 2028 [ 26 ] despite low birth rates, with the increased population resulting from inward migration [ 27 ]. Scotland’s population is becoming more ethnically diverse [ 28 ] and susceptibility to different health conditions varies by ethnic group, which has implications for the planning and provision of health services [ 29 ]. 7% of the current Scottish population are non-UK nationals and 10% were born outside Britain. The commonest countries of origin were Poland, Ireland, Italy, Nigeria and India [ 30 ].

Within Scotland, linking health data to ethnicity is standard in order to monitor and improve health of minority groups [ 31 ]. Ethnic background can differ from country of birth which means migration status cannot be assumed [ 32 ], although health inequalities experienced by migrants often extend to affect all ethnic minority groups [ 33 ]. The Scottish Health and Ethnicity Linkage Study (SHELS) linked census data to health records of 91% of the population which has provided information on mortality and morbidity by ethnic group and country of birth [ 34 ]. SHELS research indicates that the white-Scottish population have a higher mortality rate than other ethnic groups. This may be consequent to the comparatively poor health of the Scottish population relative to other European nations: high mortality rates in the general population may cause a perception that the health of minorities is more advantageous than in reality [ 35 ].

Cezard et al’s [ 13 ] analysis of self-perceived health among people in Scotland found that being born abroad had a positive impact on health status. Health declined with increased length of residence, which may be explained by cultural convergence with the majority population. Allik et al. [ 36 ] compared health inequalities by ethnic background and found that with increasing age, health differences reduced thus people aged over 75 of all ethnicities had similar or worse health status than White-Scottish people. While working-age migrants appear to be healthier than the White Scottish population, it cannot be assumed that in future this would extend to older age groups.

Research has shown deprivation as a cause of heath inequalities among ethnic minority and migrant groups [ 37 ]. The socio-economic status of minority ethnic groups in Scotland is unusual, as most are of similar or higher status than the white-Scottish population [ 38 ]. Therefore, public health interventions targeting deprivation may not address risk-factors for ethnic minorities and migrants [ 36 ]. Further research on determinants of health in migrants can help with planning and design of inclusive policies.

The 2011 census indicated that 50% of immigrants lived in the cities of Edinburgh, Glasgow, and Aberdeen. Glasgow had a greater percentage of non-European immigrants due to participation in the Asylum dispersal programme [ 39 ]. 10% of UK asylum seekers are placed in Glasgow, but records are not kept following approval of asylum claims, therefore the size of the refugee population is unknown [ 40 ]. While immigration is controlled by the British government, in policy areas devolved to the Scottish government, refugees and asylum seekers have more rights than elsewhere in UK, including access to primary healthcare for undocumented migrants [ 40 ]. Despite the mitigating effect of Scottish policies, asylum seekers’ health is worsened by the asylum process and associated poverty, marginalisation, and discrimination [ 40 ]. Health deteriorates with increasing length of time in the asylum system [ 40 ] and asylum seekers and refugees have additional health needs and require enhanced support [ 41 ]. Research on the health needs of asylum seekers in Scotland is required to ensure adequate healthcare.

Aim and objectives

While scoping reviews on migrant health have been carried out in Europe [ 12 ], Ireland [ 24 ] and the UK [ 23 ] none are currently specific to the Scottish context. Given the devolved government of Scotland and demographics described above, a targeted review would help to clarify research priorities, with the aim of improving health and health care within the migrant community in Scotland. This work therefore builds on the published scoping review of migrant health in the Republic of Ireland [ 24 ]. The authors recommend replication of the study in other countries to facilitate cross-country comparison. Our aim was to scope peer-reviewed research and grey literature on migrant health conducted in Scotland and identify any gaps in the evidence. Our objectives were to: [1] understand the extent of the available research by topic area [2] summarise the types of research already conducted, populations studied, topics covered and approaches taken [3], map the existing research conducted in Scotland and [4] identify areas for future research based on any gaps in the evidence identified.

A scoping review was conducted as they can aid detection of evidence gaps [ 42 ] and allow incorporation of grey literature in topics with insufficient published research [ 43 ]. Arksey and O’Malley’s [ 44 ] five stage scoping review framework was used.

Stage 1: identifying the research question

Arskey and O’Malley [ 44 ] suggest maintaining a broad approach to identifying the research question, in order to generate breadth of coverage. On this basis, and in line with the research question identified in the Villarroel et al. [ 24 ] scoping review, our research question was framed as follows: What is the scope, main topics and gaps in evidence in the existing literature on health of international migrants living in Scotland? Arksey and O’Malley [ 44 ] highlight the importance of defining terminology at the outset of scoping reviews. For consistency, we used the broad definition of ‘migrant’ as per Villaroel et al. [ 24 ], from the International Organisation for Migration (IOM) [ 1 ]. References to refugees or asylum seekers followed the United Nations Refugee Agency definitions [ 3 ].

Stage 2: identifying relevant studies

Electronic database searches identified reports alongside a grey literature search, in line with Arskey and O’Malley’s [ 44 ] guidance to search for evidence via different sources. CINAHL, Web of Science, SocIndex and Medline academic databases were selected with input from co-authors. Search terms for the review were based upon those used by Villaroel et al. [ 24 ] with additional relevant terms from Hannigan et al. [ 9 ] The strategy combined three sets of terms for: Migrants (e.g., refugee, migrant, immigrant or newcomer), Scotland and Health. Both free text terms and index terms were used and adapted to the 4 academic databases and searches were run on 10th March 2023 (see Additional File 1 for database search strategies). Thirteen Government, University, and third-sector websites in Scotland were scoped for selection then hand-searched for grey literature (listed in Additional File 1 ).

Stage 3: study selection

Net-migration to Scotland increased in the 2000s [ 27 ] hence a date range of January 2002-March 2023 was used to identify evidence. The search was limited to English only. Inclusion/exclusion criteria for the studies were based on those used by Villaroel et al. [ 24 ] and expanded upon following discussion with co-authors (see Table  1 ). Reports were included if based on primary or secondary research on the health of international migrants in Scotland and used qualitative, quantitative or mixed methods research design. International or UK based reports were only included if Scottish results were documented separately. Reports on the health of ethnic minority groups in Scotland was included if place of birth was recorded. Research on internal (non-international) migrants within Scotland, either moving from one Scottish area to another or from another part of the United Kingdom to Scotland, were excluded.

Stage 4: data charting

All records were saved to RefWorks for screening. Records were first screened at title/abstract stage with 10% independently checked by the co-authors. The remaining reports were single screened using full text by the first author. Data from the included records was extracted and organised in tabular form under the following headings, which were agreed by team members: article type (peer-reviewed article or grey literature), publication date, geographical setting, study/intervention’s target population, funding, primary research focus on migrant health (y/n), study objective, data collection method, study design (qualitative/quantitative/mixed) and main finding. Reports were not critically appraised in this scoping review.

Stage 5: collating, summarising and reporting results

A report (either a peer-reviewed journal article or grey literature report) is used as our unit of analysis. In order to present the range of research identified, reports were grouped by the different headings in our data charting table and the outcomes considered for relevance to our scoping review’s aim. Our Results summarise the recency, focus, study designs and funding sources of the identified research, followed by the geographical settings and whether Scotland was included in international research reports. Reports were grouped by their study population and further sub-divided by publication type and geographical area for summarising. Finally, the WHO’s European strategy and action plan (SAAP) for refugee and migrant health [ 7 ] is a policy framework designed to help governments and other stakeholders monitor and improve migrant health in Europe. There are nine strategic areas in the WHO’s SAAP, which prioritise the most salient issues. In line with Villaroel et al’s [ 24 ] approach and in order to compare scoping review outcomes, these areas were used to categorise the findings of this review. Each report was matched to the most appropriate SAAP:

Establishing a Framework for Collaborative Action.

Advocating for the right to health of refugees.

Addressing the social determinants of health.

Achieving public health preparedness and ensuring an effective response.

Strengthening health systems and their resilience.

Preventing communicable disease.

Preventing and reducing the risks caused by non-communicable disease.

Ensuring ethical and effective health screening and assessment.

Improving health information and communication.

The primary focus (aims and objectives) of each report was used to identify the relevant SAAP area/areas. To improve reliability, results were compared using coding criteria used in Villaroel et al’s study (MacFarlane 2023, personal communication, 31st May). 10% of the reports were checked by one co-author to ensure consistent coding to SAAP categories. Any instances of uncertainty in mapping reports to the relevant SAAP area/areas were discussed and resolved by team members.

This scoping review of the literature on migrant health in Scotland identified 2166 records from academic literature databases, following duplicate removal, and 170 records from website searches (see Fig.  1 ). Following screening, a total of 71 peer-reviewed journal articles and 29 grey literature studies (totalling 100 reports) were included for analysis (Results table and reference list are presented in Additional File 2 ).

Flow chart illustrating the identification of sources of evidence included in the scoping review

Overall findings

The majority of reports were published between 2013 and 2022. Fifty-eight reports (58%) focused exclusively on migrant health [ 18 , 39 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 , 66 , 67 , 68 , 69 , 70 , 71 , 72 , 73 , 74 , 75 , 76 , 77 , 78 , 79 , 80 , 81 , 82 , 83 , 84 , 85 , 86 , 87 , 88 , 89 , 90 , 91 , 92 , 93 , 94 , 95 , 96 , 97 , 98 , 99 , 100 , 101 , 102 ]. 23 centred on health but included other populations in addition to migrants – for example research on ethnic minorities or other vulnerable groups [ 13 , 31 , 35 , 103 , 104 , 105 , 106 , 107 , 108 , 109 , 110 , 111 , 112 , 113 , 114 , 115 , 116 , 117 , 118 , 119 , 120 , 121 , 122 ]. Seventeen reports were included where the sample population were migrants, but the primary topic was not health – for example destitution, integration, and service needs [ 27 , 73 , 74 , 123 , 124 , 125 , 126 , 127 , 128 , 129 , 130 , 131 , 132 , 133 , 134 , 135 ]. Health data was reported as part of the wider subject matter. One report [ 136 ] looked at the social determinants of breastfeeding including migrant status and one [ 137 ] compared attitudes to aging and family support between countries.

Funding sources were not declared for 35 (35%) of reports. The Scottish Government funded 20 reports (20%) [ 13 , 27 , 32 , 39 , 45 , 46 , 47 , 66 , 77 , 88 , 99 , 100 , 101 , 102 , 113 , 116 , 119 , 121 , 129 , 134 ]. Other common sources of funding included Government funded public bodies ( n  = 13) [ 45 , 48 , 49 , 50 , 51 , 52 , 53 , 104 , 107 , 113 , 116 , 131 , 136 ], the Scottish Health Service ( n  = 18) (either the National Health Service (NHS) [ 13 , 54 , 56 , 57 , 58 , 59 , 102 , 113 , 116 ], local NHS trusts [ 45 , 60 , 61 , 77 , 102 , 103 , 112 ] or by Public Health Scotland [ 13 , 113 ]) Eleven reports (11%) were funded by Universities. The charity sector financed 15 (15%) reports [ 53 , 63 , 66 , 69 , 70 , 71 , 72 , 73 , 74 , 103 , 111 , 123 , 125 , 132 , 138 ] and the EU and Scottish local authorities funded four reports each [ 45 , 62 , 75 , 76 , 77 , 102 , 125 , 135 ]. Professional bodies financed one report [ 126 ] as did the Japanese government [ 64 ]. No reports received funding from the business sector. The biggest sources of funding for grey literature were Refugee charities (40%) and the Scottish government (30%) (see Fig. 2 ).

Sources of funding for migrant health research in Scotland

Research methods and data collection

52% of reports used qualitative research methods. Forty-five reports (86%) collected data using 1–1 interviews and 24 (46%) used focus groups. Other methods of data collection included questionnaires (six studies (11%)), workshops (two studies (3.85%)) and observation (two studies (3.85%)). Oral/written evidence, guided play sessions, family case studies and participatory activity sessions were used in one report each.

28% of reports used quantitative research methods, most commonly cross section design (ten studies (36%)) and cohort design (18 studies (64%)). Information was obtained from databases including medical records, Census data and national records in 21 reports (75%). Questionnaires were used in six reports (21%). Other methods including body measurements, food diaries, blood samples, interviews and case reviews were used in 1 report each.

20% of reports used mixed methods. The most common method of data collection was questionnaires in 14 reports (70%), interviews in ten reports (50%), focus groups in seven reports (35%), workshops in three reports (13.6%), and databases in three reports (13.6%). Other methods included literature review in two reports (10%), case note reviews in two reports (10%) and one reports each used mapping and school records.

Geographical areas of study

Ninety-one reports were situated in Scotland, of which 35 (38.5%) covered the whole country and 56 (61.5%) specified a city or area where research was undertaken. Some UK and international reports also specified the area of Scotland. The largest share of research within Scotland overall was in Glasgow with 36 reports, followed by Edinburgh with 16 reports, Lothian with six reports, Aberdeen with five reports and Grampian with three reports. The Northeast, Stirling, Highlands, Inverness, Lanarkshire, Motherwell and Selkirk had one report in each area.

There were seven international reports, three on mortality by country of birth [ 75 , 76 , 78 ], one on cross cultural communication [ 79 ], one on maternity care in Poland and Scotland [ 99 ], one comparing attitudes to aging in China and Scotland [ 137 ] and one on the link between birthweights and integration of migrants [ 64 ]. The remaining two reports were UK based, one on immunisation of Roma and traveller communities [ 117 ] and one on the link between ethnic diversity and mortality [ 104 ]. All the included international and UK reports documented the Scottish data separately within results.

Migrant population

Thirty-one reports included all migrants in the study population. The remaining reports included 30 studies on asylum seekers/refugees, 11 on Polish migrants, ten on Africans, six each on South Asians/Chinese/European, three on Arabs, and two on Roma populations (see Fig.  3 ). Most reports did not specify the country of origin for Asylum seekers and refugees - where country of birth was specified, reports were also included in the appropriate category.

Migrant populations studied in health research in Scotland

Grey literature and peer-reviewed reports differed in population focus. The most common populations of interest in grey literature were asylum seekers/refugees consisting of 18 reports (62%) [ 27 , 47 , 54 , 55 , 59 , 63 , 70 , 71 , 72 , 73 , 74 , 123 , 125 , 127 , 128 , 132 , 134 , 138 ] while for peer-reviewed journals 24 reports (34%) focused on all migrants [ 13 , 35 , 45 , 48 , 64 , 76 , 78 , 79 , 80 , 81 , 104 , 105 , 108 , 109 , 113 , 114 , 115 , 116 , 118 , 120 , 121 , 122 , 136 ].

Migrant study population also differed by local area; Glasgow city, where the majority of research occurred, had 18 reports of 36 (50%) on Asylum seekers/refugees [ 47 , 48 , 52 , 53 , 54 , 55 , 58 , 63 , 70 , 71 , 72 , 82 , 83 , 127 , 128 , 130 , 138 , 139 ] eight reports (22%) on Africans [ 52 , 53 , 84 , 85 , 86 , 87 , 106 , 107 ], seven reports (19%) on all migrants [ 45 , 48 , 80 , 102 , 104 , 105 , 121 ] and two reports (5.5%) on Roma migrants [ 103 , 117 ]. Other populations had one reports each. In Edinburgh five reports of 16 (31%) were on the Polish population [ 56 , 67 , 68 , 89 , 90 ], and two reports (12.5%) on Asylum seekers/refugees [ 60 , 133 ], Chinese [ 62 , 137 ], South Asian [ 46 , 119 ], all migrants [ 105 , 121 ] and Africans [ 87 , 107 ]. The remaining migrant groups had one report each. Other areas of Scotland show no clear pattern with studies in disparate migrant population groups.

Number of reports per Strategic and Action Plan (SAAP) Area

SAAP Area mapping

1. establishing a framework for collaborative action.

Nine reports had a primary focus on collaborative action and were categorised under SAAP area 1 (see Fig.  4 ) [ 66 , 70 , 72 , 73 , 103 , 125 , 129 , 132 , 134 ]. Four reports (33%) used a mixed methods study design, the remaining five reports (67%) used a qualitative design. One report [ 66 ] focused on the epidemiology of female genital mutilation and a proposed intervention strategy. One report [ 66 ] focused on the epidemiology of female genital mutilation and a proposed intervention strategy. One report [ 103 ] evaluated service provision to the Roma community in Glasgow. The remaining reports focused on refugees and asylum seekers: four [ 73 , 125 , 132 , 134 ] evaluations of refugee integration projects, one [ 70 ] on services available to pregnant women, and one [ 72 ] an assessment of a peer-education service. One report [ 129 ] was a review of service provisions for migrants during the Covid-19 pandemic. All reports in SAAP area 1 were grey literature and three (37.5%) had a primary focus on migrant health while four (50%) focused on integration, one (11%) included data on ethnic minorities and one (11%) on services during the covid-19 pandemic. The majority (seven reports (78%)) were also categorised to another SAAP area most commonly area 2 (five studies (55%)) or area 5 (four studies (44%)).

2. Advocating for the right to health of refugees

Nineteen reports focused on SAAP area 2, advocating for the right to health of refugees (see Fig.  4 ) [ 47 , 52 , 53 , 54 , 55 , 63 , 70 , 71 , 83 , 103 , 123 , 124 , 125 , 127 , 128 , 129 , 134 , 138 , 140 ]. Sixteen reports (84%) had a qualitative study design and the remaining three (16%) reports used mixed methods. Nine reports (47%) focused on the health impact of the asylum system [ 52 , 55 , 71 , 74 , 123 , 127 , 128 , 129 , 138 ], five (26%) on health and access to care [ 47 , 54 , 83 , 103 , 124 ], two (10.5%) on maternity care [ 63 , 70 ], two (10.5%) on integration services [ 125 , 134 ] and one report on mental health in HIV positive migrants [ 53 ]. Nine reports (47%) had a primary focus on migrant health while the remaining 10 (53%) also involved wider social issues. The majority (15 (79%)) of reports were grey literature. All the articles in this group overlapped with another SAAP area. Area 3 is the most common joint category with ten reports (53%) followed by area 5 with seven reports (37%), area 1 shares five reports (26%), while areas 4 and 8 share one report each (5%).

3. Addressing the social determinants of health

Twenty-nine reports were categorised to SAAP area 3 – addressing the social determinants of health (see Fig.  4 ) [ 13 , 27 , 45 , 50 , 52 , 55 , 60 , 62 , 63 , 65 , 68 , 71 , 74 , 80 , 81 , 82 , 91 , 92 , 93 , 102 , 112 , 123 , 124 , 127 , 128 , 136 , 137 , 138 ]. The majority (14 (48%)) used a qualitative study method, eight (28%) used quantitative methodology and the remaining seven reports (24%) used mixed methods. Nineteen reports (65.5%) were peer-reviewed journals [ 13 , 45 , 50 , 52 , 60 , 62 , 63 , 65 , 68 , 80 , 81 , 82 , 91 , 92 , 93 , 104 , 112 , 124 , 136 , 137 ] and ten (34.5%) were grey literature [ 27 , 55 , 63 , 71 , 74 , 102 , 123 , 127 , 128 , 138 ]. Ten reports (34.5%) discussed the effects of the asylum system on health [ 27 , 52 , 63 , 71 , 74 , 123 , 124 , 127 , 128 , 137 ] and one (3.5%) migration and health [ 50 ]. Six reports (21%) focused on culture and ethnicity [ 82 , 92 , 102 , 104 , 112 , 137 ], five reports (17%) discussed economic and environmental determinants of health [ 13 , 45 , 67 , 81 , 93 ] and five reports (17%) the health impact of social activities [ 55 , 60 , 62 , 80 , 91 ]. Of the remaining reports, one [ 65 ] discussed Brexit and mental health of European migrants and one discussed the effect of coping strategies on wellbeing in Polish migrants [ 68 ]. Most reports, 18 (62%) had a primary focus on migrant health [ 45 , 50 , 52 , 55 , 60 , 62 , 63 , 65 , 67 , 68 , 71 , 80 , 81 , 82 , 91 , 92 , 93 , 102 ], six reports (21%) discussed wider social factors in addition to health [ 74 , 123 , 124 , 127 , 128 , 138 ]. Of the remaining reports three (10%) looked at ethnic background and country of birth [ 13 , 112 , 136 ], one [ 27 ] included other vulnerable groups and one [ 137 ] included people living in China and Chinese migrants to Scotland. Thirteen reports were also categorised to one or more additional SAAP area - ten (34%) were also applicable to area 2 [ 52 , 55 , 63 , 71 , 74 , 123 , 124 , 127 , 128 , 138 ], three (10%) to area 5 [ 63 , 82 , 92 ] and one (7%) to area 4 [ 27 ].

4. Achieving public health preparedness and ensuring an effective response

Twenty-one reports were assigned to SAAP area 4 (see Fig.  4 ) [ 27 , 31 , 35 , 39 , 47 , 57 , 64 , 75 , 76 , 77 , 78 , 94 , 104 , 108 , 109 , 111 , 113 , 114 , 116 , 120 , 135 ] of which fourteen (67%) used quantitative research methods, four (19%) mixed methods and three (14%) qualitative methods. Thirteen (62%) reports were peer-reviewed journals [ 35 , 59 , 64 , 75 , 78 , 104 , 108 , 109 , 111 , 113 , 114 , 116 , 120 ] and eight (38%) grey literature [ 27 , 31 , 39 , 47 , 57 , 77 , 94 , 135 ]. Most reports (12 (57%)) focused on morbidity and mortality in migrant populations [ 31 , 35 , 64 , 75 , 76 , 78 , 104 , 108 , 109 , 113 , 114 , 116 ]. Six (29%) investigated health status and healthcare needs in migrant groups in Scotland [ 39 , 47 , 57 , 77 , 94 , 135 ]. Two reports (9.5%) analysed the epidemiology of HIV infections [ 111 , 120 ] and the remaining report focused on the health needs of young people during the covid-19 pandemic [ 27 ]. Nine reports (43%) had a primary focus on migrant health [ 39 , 47 , 55 , 64 , 75 , 76 , 77 , 78 , 94 ] while eight (38%) also analysed data by ethnicity [ 31 , 35 , 104 , 108 , 109 , 113 , 114 , 116 ]. Of the remaining reports, three (14%) included other populations within Scotland [ 27 , 111 , 120 ] and one (5%) included other characteristics in addition to health information [ 135 ]. Ten reports (48%) were also categorised to another SAAP area; one to area 2 [ 47 ], one to area 3 [ 27 ], four to area 5 [ 47 , 57 , 77 , 135 ], two to area 6 [ 111 , 120 ] and two to area 9 [ 31 , 108 ].

5. Strengthening health systems and their resilience

Twenty-nine reports were assigned to SAAP area 5 (see Fig.  4 ) [ 18 , 47 , 48 , 49 , 54 , 57 , 63 , 69 , 70 , 72 , 77 , 79 , 82 , 83 , 92 , 95 , 96 , 97 , 99 , 101 , 103 , 118 , 119 , 126 , 129 , 131 , 133 , 135 , 141 ] of which 23 (79%) used qualitative research methods. Three reports used quantitative methods (10.3%) and the remaining three used mixed methods (10.3%). Twelve reports (41%) examined migrants needs and experiences of health care [ 47 , 49 , 54 , 57 , 58 , 77 , 83 , 95 , 103 , 119 , 129 , 135 ], eight (24%) focused on pregnancy and childcare [ 63 , 70 , 92 , 96 , 97 , 99 , 101 , 118 ] and two (7%) on barriers to healthcare access [ 48 , 131 ]. Two reports (7%) evaluated healthcare programmes [ 72 , 133 ] and two focused on communication in primary care [ 79 ] and maternity services [ 69 ]. The remaining three reports (10%) covered sexual health [ 82 ], health information needs of Syrian refugees [ 126 ] and general practitioner training [ 18 ]. Nineteen (65.5%) were peer reviewed journals [ 18 , 48 , 49 , 58 , 69 , 79 , 82 , 83 , 92 , 95 , 96 , 97 , 99 , 101 , 118 , 119 , 125 , 131 , 133 ] and ten (34.5%) were grey literature [ 47 , 54 , 57 , 63 , 70 , 72 , 77 , 103 , 129 , 135 ]. Twenty-one (72%) had a primary focus on migrant health [ 18 , 47 , 48 , 49 , 54 , 57 , 58 , 63 , 69 , 70 , 72 , 77 , 79 , 82 , 83 , 92 , 95 , 96 , 97 , 99 , 101 ]. Six reports (21%) included research on other characteristics or services [ 103 , 126 , 129 , 131 , 133 , 135 ]. The remaining two reports (7%) included ethnic groups as well as migrants in the data [ 118 , 119 ]. Nineteen reports (65.5%) were also assigned to one or more other category areas: five reports (17%) to area 1 [ 47 , 70 , 72 , 103 , 129 ], five reports (17%) to area 2 [ 54 , 63 , 83 , 103 , 129 ], three reports (10%) to area 3 [ 63 , 82 , 92 ], four reports (14%) to area 4 [ 47 , 57 , 77 , 135 ], one (3.5%) to area 7 [ 119 ] and one (3.5%) to area 9 [ 48 ].

6. Preventing communicable diseases

Fourteen reports were assigned to SAAP area 6 (see Fig.  4 ) [ 56 , 61 , 87 , 88 , 89 , 90 , 105 , 106 , 107 , 111 , 115 , 117 , 120 , 122 ] of which four (31%) used quantitative methods, five (38%) used qualitative methods and five (38%) used mixed methods. Five reports (38.5%) examined immunisation behaviour [ 56 , 61 , 89 , 90 , 117 ], five (38%) on epidemiology and treatment of HIV [ 106 , 107 , 111 , 120 , 122 ]. The remaining four reports (31%) focused on tuberculosis in healthcare workers [ 115 ], malaria [ 105 ] and sexual health services [ 87 , 88 ]. Only one reports was grey literature [ 88 ], the remainder were peer-reviewed journals. Six reports (46%) had a primary focus on migrant health [ 56 , 61 , 87 , 88 , 89 , 90 ] while seven reports (54%) also included other at-risk groups in the analysis. Four reports (31%) were also assigned to another SAAP category, two (15%) to area 4 [ 111 , 120 ] and two (15%) to area 8 [ 88 , 115 ].

7. Preventing and reducing the risks posed by non-communicable diseases

Eight reports were categorised to SAAP area 7 (see Fig.  4 ) [ 46 , 51 , 59 , 84 , 85 , 86 , 98 , 119 ] of which six (75%) used qualitative research methods, one (12.5%) used quantitative methods and one (12.5%) used mixed methods. Only one report (12.5%) was grey literature [ 59 ] the remaining seven reports (87.5%) were peer-reviewed journals [ 48 , 87 , 92 , 126 , 127 , 128 , 140 ]. Three reports (37.5%) focused on health behaviours [ 51 , 85 , 98 ], two (25%) on mental health, two (25%) on diabetes and one (12.5%) on chronic disease. Seven reports(87.5%) had a primary focus on migrant health [ 46 , 51 , 59 , 84 , 85 , 86 , 98 ], with the remaining report (12.5%) including ethnic minority groups [ 119 ]. One report (12.5%) was also assigned to SAAP area number 5 [ 119 ].

8. Ensuring ethical and effective health screening and assessment

There were six reports assigned to category 8 (see Fig.  4 ) [ 53 , 88 , 100 , 110 , 115 , 121 ] of which two (33%) used a quantitative research method, three (50%) used a qualitative method and one used mixed methods. One report (14%) was grey literature [ 88 ] the remaining five reports (83%) were peer reviewed journals [ 53 , 100 , 110 , 115 , 121 ]. Three reports (50%) focused on cancer screening in migrant women [ 21 , 100 , 110 ], one (17%) analysed access to HIV testing among African migrants [ 53 ], one (17%) on T.B in healthcare workers [ 72 ] and one (17%) on sexual health [ 36 ]. Three reports (50%) had a primary focus on migrant health [ 53 , 88 , 100 ] while the remaining three reports (50%) included other at-risk groups in the analysis [ 110 , 115 , 121 ]. There were three reports which overlapped with other SAAP areas: one [ 53 ] (17%) was categorised to area 2 while two [ 88 , 115 ] (33%) were categorised to area 6.

9. Improving health information and communication

Three reports were assigned to SAAP area 9 (see Fig.  4 ) [ 31 , 108 , 130 ]. One of these (33%) used a qualitative approach, one (33%) used a quantitative approach and one (33%) used mixed methods. Two [ 108 , 130 ] (66%) were peer-reviewed journal articles and one [ 31 ] (33%) was grey literature. Two reports (66%) focused on improving migrant demographics and health information using databases [ 31 , 108 ] while one (33%) described an information-needs matrix for refugees and asylum seekers [ 130 ]. Two [ 31 , 108 ] included ethnicities in the data while one [ 130 ] had a primary focus on migrant health. Two reports [ 31 , 108 ] (66%) also applied to SAAP area 4 while one report [ 130 ] (33%) was in SAAP area 9 only.

To our knowledge this is the first scoping review conducted on migrant health in Scotland. A previous rapid literature review [ 94 ] found most research focused on health behaviours, mental health, communicable disease and use of and access to healthcare; however, the review limited migrant definition to those who had immigrated within five years and asylum seekers were not included.

In our review, the majority of reports were published from 2013 onwards, aligning with the expansion in migrant research internationally [ 142 ]. 52% used qualitative research methods, 28% used quantitative methods and 20% used mixed methods. 58% focused on migrant health: the remaining papers included other populations or health as part of a wider remit. Research funding was mostly provided by the Scottish Government, NHS, refugee charities and Universities. No studies received funding from the private sector, although this sector has the potential resource and capacity to play a key role in funding future research to improve migrant health in Scotland. Geographically, most studies took place in Glasgow (36%), nationwide (38.5%) or Edinburgh (16%) – other areas were under-represented including Aberdeen (5%), despite being the city with the largest migrant population [ 30 ]. There was a lack of studies in rural localities. These findings concur with a UK migrant health review by Burns et al. [ 23 ] where research was concentrated in larger cities and data was sparse in rural areas relative to the migrant population.

Half of the research identified that was conducted in Glasgow focused on asylum seekers/refugees. Glasgow was previously the only Scottish city to host asylum seekers [ 143 ] and currently supports the most asylum seekers of any local authority in the UK [ 29 ]. In April 2022, the UK government widened the Asylum dispersal scheme to all local authorities [ 144 ]. Around 70% of Scotland’s refugee support services are based in Glasgow and the South-west [ 145 ]. As reduced access to services may impact the health of asylum seekers, research in Glasgow may not be generalizable to other regions of Scotland.

Almost one-third (30%) of all reports focused on asylum seekers and refugees – an overrepresentation given that only 18% of migrants to the UK are asylum seekers [ 146 ] and as low as 2% of all migrants in Scotland [ 147 ]. Asylum seekers and refugees are at risk of poor health due to trauma, difficult journeys, overcrowded camps, poor nutrition and lack of access to healthcare [ 148 ]. They have worse maternity outcomes and increased rates of mental illness [ 149 ]. Increased research on health of asylum seekers and refugees is necessary due to their additional vulnerabilities [ 142 ]. However, asylum seeker’s country of origin was generally not specified. Asylum seekers have heterogenic backgrounds [ 150 ] and nationality and trauma experience affect health status [ 151 ]. Further research focused on specific nationalities of asylum seekers would enhance understanding of the health needs in this population.

Almost one-third (31%) of studies did not specify a migrant group. This concurs with a Norwegian migrant health study by Laue et al. [ 152 ] where 36% of research did not identify country of birth. Where nationality was identified, Polish, African and South Asian were most prevalent. Poles are the largest migrant group in Scotland, however for the other most common immigrant groups of Irish, Italian and Nigerian [ 30 ] there was an absence of research. No studies took place on Nigerian migrants – nine studies indicated African populations, but country of birth was not specified. Since March 2022, 23,000 Ukrainians have migrated to Scotland [ 153 ], however no studies on Ukrainians were identified currently. Research may be underway which is yet to be published.

Only one study explored the impact of Brexit on European migrants’ health despite 56% of migrants to Scotland being EU nationals [ 30 ]. Again, research may be taking place currently, which is yet to be published. No studies involved undocumented migrants despite this populations’ high rates of poor physical/mental health exacerbated by poor housing and working conditions [ 154 ]. An estimated 7.2–9.5% of the workforce in the UK are migrant workers who have higher risks of poor working conditions and injury [ 155 ]. Scotland depends on a migrant workforce for some industries such as agriculture [ 156 ] but only two research papers specified migrant workers.

Most research papers related to the right to health of refugees (SAAP 2), social determinants of health (SAAP 3), public health planning (SAAP 4) and strengthening health systems (SAAP 5). Areas with less research were frameworks for collaborative action (SAAP 1), preventing communicable disease (SAAP 6), preventing non-communicable disease (SAAP 7) and health screening and assessment (SAAP 8). Only three studies related to improving health information and communication (SAAP 9). Lebano et al. [ 12 ] conducted a literature review of migrant health in Europe and found data collection unreliable and disorganised. There is a lack of data on the numbers and types of migrants entering Scotland and research tends not to differentiate between ethnic minorities and migrants [ 94 ]. As poor-quality information hinders surveillance and planning of services SAAP area 9 is an important consideration for increased research.

Villarroel et al. [ 24 ] also found more research in SAAP areas 3 to 5 and less in areas 6 to 9. However, their study returned no results in category 1, collaborative action, or 2, the right to health of refugees, while this study assigned 9% of articles to category 1 and 19% to category 2. Most articles in our study relating to categories 1 and 2 were grey literature, which was excluded from the original Irish scoping review. This highlights a potential difference in the focus of peer-reviewed articles compared to government/refugee charity commissioned reports. Collaborative action and the right to health of refugees and asylum seekers are entwined in Scotland due to the complex policy environment; the social determinants of health such as housing, education, welfare rights and social integration are influenced by a variety of UK and Scottish statutory bodies as well as third sector organisations [ 157 ]. Despite this complexity, organisations work well together [ 158 ]. Further academic research in this area would enhance joint working practices and networks.

A scoping review in the UK [ 23 ] found similar quantities of research corresponding to SAAP areas 3, 2 and 9. However in Scotland areas 1, 5 and 8 were a combined 44% of included papers compared with 27.8% of results on health systems and structures in Burns et al’s [ 23 ] study. Almost half of the articles in SAAP areas 1,5 and 8 were grey literature, which was not included in Burns et al’s [ 23 ] review. Conversely, Burns et al. [ 23 ] found 81.9% of research in the UK related to epidemiology, equivalent to SAAP categories 4,6 and 7. In a Norwegian scoping review of migrant health [ 152 ] 65% of research was related to epidemiological data on health and disease. Only 42% of the research in this current study related to epidemiological data; the quantity of evidence was reduced by excluding combined research from the UK. As Scotland has higher mortality and morbidity than elsewhere in the UK [ 29 ] it is important to undertake further epidemiological research limited to Scotland.

Strengths and weaknesses

Strengths of this review include the use of the WHO’s SAAP categories [ 7 ] to classify data, in accordance with the Villarroel et al’s [ 24 ] study: this means results are linked to policy on migrant health and facilitates comparability to the Irish study results. Additionally results include data on migrant groups, locality, and funding of included papers; these highlight potential omissions for future research consideration. Results include diverse research methods and published and grey literature giving a wide overview of available evidence, reported using the Preferred Reporting Items for Systematic reviews and Meta-Analyses for Scoping Reviews (PRISMA-ScR) checklist (see Additional File 3 ) [ 159 ].

Limitations included the lack of an open-access protocol and search limitations of English language and selected databases. This means some relevant reports may be omitted. Due to time and resource limitations no quality appraisal was planned for included reports. Whilst we did not synthesise the findings for each topic area and migrant group, future systematic reviews could be undertaken to address this limitation and build on this work.

Conclusions

Immigration and ethnic diversity in Scotland have increased since 2002 which is reflected in the expansion of migrant health research. This review highlights evidence gaps including a lack of research in rural areas, undocumented migrants and migrant workers. There is a tendency to cluster asylum seekers together rather than differentiate between national groups. Within the SAAP areas there is less evidence relating to collaborative action, preventing communicable disease, preventing non-communicable disease and health screening and assessment. Further research is required on improving health information and communication for migrant populations in Scotland – a significant omission given the importance of accurate information for health service planning.

Availability of data and materials

All data analysed during this review comes from the papers listed in Additional file 2 .

Abbreviations

European Union

Human Immunodeficiency Virus

National Health Service

Strategy and Action Plan

The Scottish Health and Ethnicity Linkage Study

United Kingdom

World Health Organisation

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Acknowledgements

Thank-you to Professor Anne MacFarlane and PHD student Anne Cronin, of the University of Limerick, Ireland for sharing the coding guidelines currently used in an update to Villarroel et. al’s 2019 study on Migrant Health in the Republic of Ireland.

No funding was received for this work, which was undertaken as G. Petrie’s Master of Public Health dissertation module at the University of Stirling.

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KA, RO and GP finalised the study design collectively. GP conducted the searches, analysis and write up, with support from KA and RO. All three authors read and approved the manuscript prior to submission.

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Correspondence to R. O’Donnell .

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Petrie, G., Angus, K. & O’Donnell, R. A scoping review of academic and grey literature on migrant health research conducted in Scotland. BMC Public Health 24 , 1156 (2024). https://doi.org/10.1186/s12889-024-18628-1

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The Literature Review: A Foundation for High-Quality Medical Education Research

a  These are subscription resources. Researchers should check with their librarian to determine their access rights.

Despite a surge in published scholarship in medical education 1 and rapid growth in journals that publish educational research, manuscript acceptance rates continue to fall. 2 Failure to conduct a thorough, accurate, and up-to-date literature review identifying an important problem and placing the study in context is consistently identified as one of the top reasons for rejection. 3 , 4 The purpose of this editorial is to provide a road map and practical recommendations for planning a literature review. By understanding the goals of a literature review and following a few basic processes, authors can enhance both the quality of their educational research and the likelihood of publication in the Journal of Graduate Medical Education ( JGME ) and in other journals.

The Literature Review Defined

In medical education, no organization has articulated a formal definition of a literature review for a research paper; thus, a literature review can take a number of forms. Depending on the type of article, target journal, and specific topic, these forms will vary in methodology, rigor, and depth. Several organizations have published guidelines for conducting an intensive literature search intended for formal systematic reviews, both broadly (eg, PRISMA) 5 and within medical education, 6 and there are excellent commentaries to guide authors of systematic reviews. 7 , 8

• A literature review forms the basis for high-quality medical education research and helps maximize relevance, originality, generalizability, and impact.
• A literature review provides context, informs methodology, maximizes innovation, avoids duplicative research, and ensures that professional standards are met.
• Literature reviews take time, are iterative, and should continue throughout the research process.
• Researchers should maximize the use of human resources (librarians, colleagues), search tools (databases/search engines), and existing literature (related articles).
• Keeping organized is critical.

Such work is outside the scope of this article, which focuses on literature reviews to inform reports of original medical education research. We define such a literature review as a synthetic review and summary of what is known and unknown regarding the topic of a scholarly body of work, including the current work's place within the existing knowledge . While this type of literature review may not require the intensive search processes mandated by systematic reviews, it merits a thoughtful and rigorous approach.

Purpose and Importance of the Literature Review

An understanding of the current literature is critical for all phases of a research study. Lingard 9 recently invoked the “journal-as-conversation” metaphor as a way of understanding how one's research fits into the larger medical education conversation. As she described it: “Imagine yourself joining a conversation at a social event. After you hang about eavesdropping to get the drift of what's being said (the conversational equivalent of the literature review), you join the conversation with a contribution that signals your shared interest in the topic, your knowledge of what's already been said, and your intention.” 9

The literature review helps any researcher “join the conversation” by providing context, informing methodology, identifying innovation, minimizing duplicative research, and ensuring that professional standards are met. Understanding the current literature also promotes scholarship, as proposed by Boyer, 10 by contributing to 5 of the 6 standards by which scholarly work should be evaluated. 11 Specifically, the review helps the researcher (1) articulate clear goals, (2) show evidence of adequate preparation, (3) select appropriate methods, (4) communicate relevant results, and (5) engage in reflective critique.

Failure to conduct a high-quality literature review is associated with several problems identified in the medical education literature, including studies that are repetitive, not grounded in theory, methodologically weak, and fail to expand knowledge beyond a single setting. 12 Indeed, medical education scholars complain that many studies repeat work already published and contribute little new knowledge—a likely cause of which is failure to conduct a proper literature review. 3 , 4

Likewise, studies that lack theoretical grounding or a conceptual framework make study design and interpretation difficult. 13 When theory is used in medical education studies, it is often invoked at a superficial level. As Norman 14 noted, when theory is used appropriately, it helps articulate variables that might be linked together and why, and it allows the researcher to make hypotheses and define a study's context and scope. Ultimately, a proper literature review is a first critical step toward identifying relevant conceptual frameworks.

Another problem is that many medical education studies are methodologically weak. 12 Good research requires trained investigators who can articulate relevant research questions, operationally define variables of interest, and choose the best method for specific research questions. Conducting a proper literature review helps both novice and experienced researchers select rigorous research methodologies.

Finally, many studies in medical education are “one-offs,” that is, single studies undertaken because the opportunity presented itself locally. Such studies frequently are not oriented toward progressive knowledge building and generalization to other settings. A firm grasp of the literature can encourage a programmatic approach to research.

Approaching the Literature Review

Considering these issues, journals have a responsibility to demand from authors a thoughtful synthesis of their study's position within the field, and it is the authors' responsibility to provide such a synthesis, based on a literature review. The aforementioned purposes of the literature review mandate that the review occurs throughout all phases of a study, from conception and design, to implementation and analysis, to manuscript preparation and submission.

Planning the literature review requires understanding of journal requirements, which vary greatly by journal ( table 1 ). Authors are advised to take note of common problems with reporting results of the literature review. Table 2 lists the most common problems that we have encountered as authors, reviewers, and editors.

Sample of Journals' Author Instructions for Literature Reviews Conducted as Part of Original Research Article a

Common Problem Areas for Reporting Literature Reviews in the Context of Scholarly Articles

Locating and Organizing the Literature

Three resources may facilitate identifying relevant literature: human resources, search tools, and related literature. As the process requires time, it is important to begin searching for literature early in the process (ie, the study design phase). Identifying and understanding relevant studies will increase the likelihood of designing a relevant, adaptable, generalizable, and novel study that is based on educational or learning theory and can maximize impact.

Human Resources

A medical librarian can help translate research interests into an effective search strategy, familiarize researchers with available information resources, provide information on organizing information, and introduce strategies for keeping current with emerging research. Often, librarians are also aware of research across their institutions and may be able to connect researchers with similar interests. Reaching out to colleagues for suggestions may help researchers quickly locate resources that would not otherwise be on their radar.

During this process, researchers will likely identify other researchers writing on aspects of their topic. Researchers should consider searching for the publications of these relevant researchers (see table 3 for search strategies). Additionally, institutional websites may include curriculum vitae of such relevant faculty with access to their entire publication record, including difficult to locate publications, such as book chapters, dissertations, and technical reports.

Strategies for Finding Related Researcher Publications in Databases and Search Engines

Search Tools and Related Literature

Researchers will locate the majority of needed information using databases and search engines. Excellent resources are available to guide researchers in the mechanics of literature searches. 15 , 16

Because medical education research draws on a variety of disciplines, researchers should include search tools with coverage beyond medicine (eg, psychology, nursing, education, and anthropology) and that cover several publication types, such as reports, standards, conference abstracts, and book chapters (see the box for several information resources). Many search tools include options for viewing citations of selected articles. Examining cited references provides additional articles for review and a sense of the influence of the selected article on its field.

Box Information Resources

• Web of Science a
• Education Resource Information Center (ERIC)
• Cumulative Index of Nursing & Allied Health (CINAHL) a
• Google Scholar

Once relevant articles are located, it is useful to mine those articles for additional citations. One strategy is to examine references of key articles, especially review articles, for relevant citations.

Getting Organized

As the aforementioned resources will likely provide a tremendous amount of information, organization is crucial. Researchers should determine which details are most important to their study (eg, participants, setting, methods, and outcomes) and generate a strategy for keeping those details organized and accessible. Increasingly, researchers utilize digital tools, such as Evernote, to capture such information, which enables accessibility across digital workspaces and search capabilities. Use of citation managers can also be helpful as they store citations and, in some cases, can generate bibliographies ( table 4 ).

Citation Managers

Knowing When to Say When

Researchers often ask how to know when they have located enough citations. Unfortunately, there is no magic or ideal number of citations to collect. One strategy for checking coverage of the literature is to inspect references of relevant articles. As researchers review references they will start noticing a repetition of the same articles with few new articles appearing. This can indicate that the researcher has covered the literature base on a particular topic.

Putting It All Together

In preparing to write a research paper, it is important to consider which citations to include and how they will inform the introduction and discussion sections. The “Instructions to Authors” for the targeted journal will often provide guidance on structuring the literature review (or introduction) and the number of total citations permitted for each article category. Reviewing articles of similar type published in the targeted journal can also provide guidance regarding structure and average lengths of the introduction and discussion sections.

When selecting references for the introduction consider those that illustrate core background theoretical and methodological concepts, as well as recent relevant studies. The introduction should be brief and present references not as a laundry list or narrative of available literature, but rather as a synthesized summary to provide context for the current study and to identify the gap in the literature that the study intends to fill. For the discussion, citations should be thoughtfully selected to compare and contrast the present study's findings with the current literature and to indicate how the present study moves the field forward.

To facilitate writing a literature review, journals are increasingly providing helpful features to guide authors. For example, the resources available through JGME include several articles on writing. 17 The journal Perspectives on Medical Education recently launched “The Writer's Craft,” which is intended to help medical educators improve their writing. Additionally, many institutions have writing centers that provide web-based materials on writing a literature review, and some even have writing coaches.

The literature review is a vital part of medical education research and should occur throughout the research process to help researchers design a strong study and effectively communicate study results and importance. To achieve these goals, researchers are advised to plan and execute the literature review carefully. The guidance in this editorial provides considerations and recommendations that may improve the quality of literature reviews.

MINI REVIEW article

Psychological and social consequences of deafblindness for siblings: a systematic literature review.

• 1 Centre de Recherche Psychanalyse, Médecine et Société, Université Paris Cité, Paris, France
• 2 Centre de Recherche en Psychopathologie et Psychologie Clinique, Université Lumière Lyon 2, Lyon, France

The onset of deafblindness profoundly impacts both the individual with this condition and the individual’s family, including siblings. While current studies have primarily focused on the impact felt by parents or spouses, the distinct experiences of siblings have received comparatively less attention. This systematic review addresses the existing research gap regarding the psychological and social consequences experienced by siblings of individuals with deafblindness. A comprehensive search was conducted across multiple electronic databases, including PsycINFO, PsycARTICLES, Dissertations & Theses (on ProQuest), ERIC (Education Resources Information Center), International Bibliography of the Social Sciences (IBSS), Sociological Abstracts, Google Scholar, PubMed, and Cairn Info. Seven studies were identified as meeting the eligibility criteria for inclusion. The review revealed that siblings of individuals with deafblindness face psychological and social challenges, including emotions such as feelings of neglect, resentment, embarrassment, jealousy, and anxiety. Siblings also grapple with communication difficulties, contributing to feelings of exclusion and insecurity. In addition, these siblings take on significant responsibilities within the family and encounter obstacles in forming relationships outside the family. These findings underscore the need of interventions to improve the well-being of siblings of individuals with deafblindness by addressing their psycho-emotional needs and promoting positive social interactions. These findings align with studies conducted on siblings of children with other disabilities. However, additional research is crucial to investigate overlooked dimensions, particularly positive factors like coping mechanisms and resilience, that may influence the mental health and social experiences of these siblings.

Introduction

According to Watters et al. (2005) , deafblindness is “a condition that combines any degree of hearing loss with any degree of vision loss that interferes with communicating and acquiring information, even though individuals who are deafblind may still have varying levels of useful vision and hearing” (p. 16). Indeed, deafblindness encompasses a spectrum of manifestations ( Rodgers, 2021 ), and most subjects exhibit residual vision and hearing. The first global report of the World Federation of the DeafBlind indicates that approximately 0.2% of the world’s population lives with severe deafblindness, whereas “milder forms” of deafblindness impact around 2% of the global population ( International Disability Alliance, 2018 ). Moreover, individuals with deafblindness frequently exhibit additional physical and cognitive impairments ( Heller et al., 1999 ).

Deafblindness manifests in various forms, including congenital, acquired, and aged-related deafblindness, each with distinct etiological factors and characteristics ( Souriau, 2000 ; Dammeyer, 2010 ). Congenital deafblindness occurs at birth or shortly thereafter ( Dammeyer, 2010 ) and is caused by genetic factors (e.g.: CHARGE syndrome), prenatal infections (e.g: cytomegalovirus, rubella,), or birth complications (e.g.: as prematurity, low birth weight,). Metabolic disorders, congenital malformations, certain medications, or maternal drug use are other potential causes of congenital deafblindness ( Chen, 2004 ; Holte et al., 2006 ; National Center on Deaf-Blindness, n.d. ).

Acquired deafblindness occurs later in life (following a period of normal sensory functioning) ( Dammeyer, 2010 ) and can be attributed to genetic disorders (e.g.: Usher syndrome), traumatic events (e.g.: severe head injuries), infections (e.g.: meningitis), or specific neuronal conditions (e.g.: multiple sclerosis). Additionally, prolonged use of certain medications or exposure to toxic substances like chemotherapy drugs, antibiotics, or environmental toxins can potentially lead to acquired deafblindness ( Chen, 2004 ; Holte et al., 2006 ; National Center on Deaf-Blindness, n.d. ).

Age-related deafblindness is characterized by a gradual decline of both hearing and vision that arises due to the natural process of aging. This condition predominantly affects older adults, typically occurring after the age of 65. The primary causes of age-related deafblindness include presbycusis (age-related hearing loss), age-related macular degeneration, cataracts, and glaucoma, in conjunction with age-related hearing loss. Furthermore, other age-related health conditions like diabetes, cardiovascular diseases, and neurological disorders can also contribute to hearing loss and vision loss ( Simcock, 2017 ).

Among syndromes associated with deafblindness, CHARGE syndrome is the most frequently encountered. However, in the context of acquired deafblindness, Usher syndrome predominates as the leading cause ( Chen, 2004 ; Holte et al., 2006 ; National Center on Deaf-Blindness, n.d. ). The impact of deafblindness, a distinct condition, is multiplicative (i.e., not simply the sum of vision and hearing impairments) ( Fletcher and Guthrie, 2013 ; Ferrell et al., 2014 ). For example, individuals with deafblindness cannot compensate for hearing loss through lip reading. Thus, deafblindness is best understood as a precise and individual condition with disabilities distinct from those associated with only vision or hearing impairment ( Arcous et al., 2019 ).

Deafblindness correlates with lower education, increased poverty, and higher unemployment rates ( International Disability Alliance, 2018 ). The age at which the condition starts has a big effect on how difficult it is to cope with, especially when the condition is present from birth, leading to significant limitations ( Bruce, 2005 ; Ronnberg and Borg, 2011 ). Progressive conditions, like Usher syndrome, bring multifaceted challenges, including access to information, mobility problems, workplace and educational difficulties, social isolation, feelings of insecurity, difficulties in projecting into the future and mental health issues ( Arcous et al., 2019 ). Despite these challenges, appropriate accommodations can mitigate difficulties, which thereby enhances the quality of life for individuals with Usher syndrome ( Ellis and Hodges, 2013 ; Arcous et al., 2019 ). These accommodations include alternative communication methods, assistive technologies, leisure activities, and social support (given by friends or family) ( Arcous et al., 2019 ).

Indeed, the family plays a fundamental role in the well-being of the person with deafblindness ( Spring et al., 2012 ). The family is the primary source of support ( Ellis and Hodges, 2013 ; Arndt and Parker, 2016 ). According to Bernard (2013) , “The quality of life of deafblind children and adults is greatly influenced by the connection, appropriate support, and interactions with the family” (p. 135). Family members can help with travel, administrative procedures, and daily tasks ( Kyle and Barnett, 2012 ). Family members also play an important role in interpreting and accessing information from the outside world ( Kyle and Barnett, 2012 ; Simcock, 2017 ). Moreover, the family offers the possibility of social openness ( Gullacksen et al., 2011 ; Arndt and Parker, 2016 ). Sometimes family members are the only people individuals with deafblindness see during the week. The family can also support self-determination of the deafblind individual ( Morgan et al., 2002 ).

According to Siemon (1984) , “Because families are a system, distress in one member affects both the system and each member in it.” (p. 294). It is necessary to acknowledge that the presence of a child with a disability in a family can lead to emotional consequences (felt among all family members), along with additional challenges like increased financial responsibilities and the need for extended caregiving. Families may also experience feelings of grief and loss, which can be amplified based on the severity of the disability ( Correa-Torres, 2008 ). Family members may experience both social and psychological consequences due to the presence of a child with a disability.

Social consequences pertain to the effects and repercussions on individuals’ social interactions, relationships, and integration within society, stemming from particular circumstances, events, or conditions. These consequences encompass strained social dynamics, social exclusion or isolation, challenges in forming and maintaining relationships, stigma or discrimination, and difficulties in engaging in social activities or fulfilling social roles ( Brooks, 1980 ; Mormiche and Boissonnat, 2003 ). Psychological consequences encompass the effects and influences on individuals’ emotional, cognitive, and behavioral well-being from experienced circumstances or events. These consequences encompass emotional responses such as stress, anxiety, depression, anger, sadness and cognitive changes ( Jover, 2014 ; Ha Namkung and Carr, 2020 ).

Regarding the social consequences, the presence of an individual living with deafblindness poses communication challenges within the family ( Souriau, 2000 ; Rodgers, 2021 ). Indeed, a study conducted by Kyle and Barnett (2012) , among 39 individuals with acquired deafblindness, demonstrated that family members may encounter difficulties acquiring tactile sign language or other specific communication skills tailored to the disability. It is thus evident that effective communication within the family plays a pivotal role in managing the disability, fostering the self-esteem of the individual with a disability, and promoting family cohesion ( Miner, 1995 ; Wahlqvist et al., 2020 ).

When deafblindness is acquired progressively, this process can provoke significant familial disruptions, which in many cases leads to entire role realignments assumed by family members ( Gullacksen et al., 2011 )— a process that is neither straightforward nor undemanding. For instance, according to Watters-Miles (2014) , who conducted a study among six individuals with Usher syndrome, parents may become overprotective of their children. Hersh (2013) study on individuals with deafblindness (unspecified type) similarly observes protective behaviors demonstrated by parents. Miner (1995) study, focusing on individuals with Usher syndrome type I, emphasizes the need for adjustments in familial relationships as the syndrome progresses. Similarly, Figueiredo et al. (2013) study, involving eleven individuals with Usher syndrome (type I or II), supports Miner’s study results.

The presence of a child with deafblindness can moreover present challenges in socialization beyond the immediate household for family members. A study conducted by Hersh (2013) , which examined twenty-seven individuals with deafblindness (without specification of the type), revealed that families with children with deafblindness might experience feelings of stigmatization and embarrassment, ultimately leading to adverse consequences for their social participation.

Regarding the psychological consequences, Watters-Miles (2014) emphasizes that the onset of a disability can be traumatic for the individual and the entire family. Miner (1995) , focusing on individuals with Usher syndrome type I, highlights that communication difficulties, hindered by the nature of the disability, can result in additional stress and can increased risk of depression among family members. Indeed, according to Correa-Torres (2008) , sighted-hearing parents of children with deafblindness are at a higher risk of developing depression. Hartshorne and Schmittel (2016) who conducted a study on the social–emotional development of children with deafblindness (all types) also found that the risk of sighted-hearing parents developing depression is greater. Emotional problems were also reported by partners of individuals with Usher type 1. These partners appear to have more daily responsibilities and feel tired, depressed and resentful ( Miner, 1995 ).

In summary, the familial implications of deafblindness are both pervasive and multidimensional, extending to siblings who have been relatively overlooked in current literature. Addressing these gaps is vital for the inclusive support and well-being of all family members. Indeed, the siblings of children with deafblindness confront unique challenges due to the very nature of their brother or sister’s disability. Because of visual and auditory impairment, siblings are greatly hindered in their ability to communicate and play with their disabled sibling.

Siblings also assume increasingly vital roles within the family, often enduring lifelong relationships with their disabled siblings ( Cicirelli, 1995 ). Indeed, siblings share a deep and enduring history together. As parents age, siblings may take on greater responsibility and play a more meaningful role in their relationship with a sibling with a disability ( Vert et al., 2016 ). Also, siblings serve as primary witnesses to the challenges faced by the disabled child and their parents ( Dayan and Scelles, 2017 ).

Despite this, research has primarily concentrated on parents and spouses, leaving a gap in understanding the psychological and social experiences of siblings of children with deafblindness. 1 To bridge this gap in knowledge, this systematic review aims to explore the existing literature on the psychological and social consequences of deafblindness on siblings. Gaining a comprehensive understanding of their experiences is crucial for providing inclusive support and enhancing the overall well-being of the entire family.

Methodology

This systematic literature review was carried out to explore the existing literature on the psychological and social consequences of deafblindness on siblings.

Search strategy

We conducted a comprehensive literature search using various electronic databases, including PsycINFO, PsycARTICLES, Dissertations & Theses (on ProQuest), ERIC (Education Resources Information Center), International Bibliography of the Social Sciences (IBSS), Sociological Abstracts, Google Scholar, PubMed, and Cairn Info. The following combination of words was used to conduct the systematic review search: “Siblings” OR “Brother,” OR “Brothers” OR “Sister” OR “Sisters” OR “Fratrie” OR “Frère” OR “frères” OR “sœurs” OR “Soeur” AND (“Sourdaveugle” OR “Sourd-aveugle” OR “Surdicécité” OR “Deaf-blind” OR “Deafblind” OR “Deafblindness” OR “Dual Sensory loss” OR “dual sensory impair” OR “Dual sensory impairment” or “Dual sensory disability” OR “Usher Syndrome” OR “Wolfram Syndrome” OR “Stickler Syndrome” OR “Charge Syndrome” OR “Alport Syndrome” OR “Bardet-Biedl Syndrome” OR “Rubella” OR “Cockayne Syndrome” OR “Cornelia de Lange Syndrome” OR “Flynn-Aird Syndrome” OR “Goldenhar Syndrome” OR “Deaf-blind Hypopigmentation Syndrome”).

Inclusion and exclusion criteria

Studies were included in the review if they were (1) written in English or in French languages (two languages spoken by the authors), (2) published in a peer-reviewed journal/ published dissertation/thesis studies or published reports with no date limits for the publication year (3) explored life experience and/or psychological health and/or social experience of siblings of individuals with deafblindness, and (4) articles mentioning the terms “fratrie” (siblings), “siblings,” “frères” (brothers), “frère” (brother), “sœurs” (sisters), “sœur” (sister).

Studies were excluded from the review if they primarily focused on age-related deafblindness. This exclusion criterion was implemented to ensure that the included studies specifically addressed the experiences of siblings who grew up in the same household as individuals with deafblindness during childhood or adolescence.

Date selection and collection process

The articles selection process was conducted by two authors (first and second author). Here are the 6 steps that we followed for the article selection: (1) Database Retrieval; (2) Duplicate Removal; (3) Title and Abstract Screening; (4) Accessibility Check; (5) Criteria-Based Selection; and (6) Final Selection.

1. Number of articles obtained in each database: the number of articles collected from each database was recorded to determine the initial pool of resources available for analysis. One thousand six hundred fourteen (1614) articles were found in total in the databases.

2. Number of articles set discarded as duplicates: to ensure data integrity and avoid redundancy, duplicate articles across databases were identified and removed from the pool. This step helped streamline the analysis process and eliminate any repetition. Two hundred and seventeen duplicates were identified, and 1,397 articles remained after removing these duplicates.

3. Number of articles set aside based on titles and abstract: after obtaining the articles (1,397 after removing the duplicates), a screening process was conducted based on their titles and abstracts. Articles that did not appear relevant to the research topic or did not meet the inclusion criteria were discarded. Many articles discussed the chosen syndromes or the chosen syndromes in relation to siblings. However, most of these studies had a medical perspective, aiming to uncover the genetic factors behind disease transmission rather than exploring the personal and social experiences of the siblings. One thousand three hundred fifty-one (1351) were thus rejected at this step in our methodology. Eighteen articles remained.

4. Among the eighteen remaining articles, we excluded some due to accessibility issues. In cases where we could only access the title or the abstract without the full text, we left these articles out of our analysis. This was done to ensure a thorough analysis, focusing on complete articles rather than just titles or abstracts. Specifically, we excluded two articles because we could only retrieve their titles, and four more because we had access only to their abstracts.

5. Number of articles selected for analysis: from the remaining articles, a selection was made based on the predetermined criteria for inclusion. These criteria could include relevance to the research question or methodology problems. Twelve articles were screened for eligibility using the inclusion and exclusion criteria. Five articles were excluded.

6. Final number of articles selected: after screening and applying the inclusion criteria, the remaining articles constituted the final set of resources selected for analysis. The number of articles (7) at this stage represents the data used in the research study.

Data extraction

The two authors extracted the selected articles based on their relevance to the inclusion criteria. One author was responsible for categorizing the results into psychological and social consequences, utilizing the definitions provided in the introduction. The second author validated this categorization. Information about the country the research was carried out, the age of the siblings, the type of deafblindness of the siblings, the methodology used in the articles and the bias and limitations presented by the authors of each article were also gathered ( Figure 1 ).

Figure 1 . Data selection process.

General characteristics of the studies

In total, there were five studies conducted in the United States, 1 study in Australia, 1 study in Canada, and 1 study in the United Kingdom. Only four studies directly assessed siblings’ experiences with their siblings with deafblindness. The other three studies examined siblings’ experiences through the perspectives of parents or individuals living with deafblindness.

Rowan (1990) conducted a study in the United States involving 12 siblings of deafblind children from five families. The age range of the siblings in this study was between 7 and 16 years old. Another study in the United States by Laman (2006) focused on four siblings ranging in age from 19 to 22 years old, with a sibling with congenital deafblindness. Harland and Cuskelly (2000) conducted a study in Australia with four siblings from different families. The age range of the siblings in this study was between 21 and 30 years old.

The causes of deafblindness in siblings with this disability were different. We believe it is necessary to clarify this because the experiences of non-disabled siblings may vary depending on the severity of the visual and hearing impairment. Rowan (1990) discussed different conditions, such as cortical visual impairment, profound hearing loss, and associated complications. Laman (2006) focused on congenital deafblindness without specifying the cause. Harland and Cuskelly (2000) explored cases of moderate to profound vision and hearing impairments, often accompanied by intellectual and physical disabilities. Vert et al. (2016) specifically studied individuals with CHARGE syndrome. Watters et al. (2005) investigated both acquired and congenital forms of deafblindness. Ellis and Hodges (2013) examined Usher syndrome, distinguishing between type I, type II, and type III. Finally, Heller et al. (1999) study focused on children with deafblindness without specifiying the various types.

Several studies have adopted qualitative methods to explore siblings’ experiences in the context of deafblindness. Rowan (1990) , a graduate student in education, conducted one-on-one interviews with siblings to gain insights into their perspectives. Laman (2006) , a doctor in special education, employed semi-structured interviews to examine adult siblings’ perceptions of their involvement in the Individualized Transition Plan (ITP) of siblings with congenital deafblindness. Harland and Cuskelly (2000) , two researchers in special education, conducted semi-structured interviews on sibling responsibilities, support, personal development, advocacy, and more topics. Watters et al. (2005) , researchers in disability studies, conducted focus groups with individuals with deafblindness, parents/advocates, and interviews with service providers. Ellis and Hodges (2013) , researchers in education, employed semi-structured and extensive interviews.

In addition to qualitative approaches, quantitative studies have examined siblings’ experiences with deafblindness. Vert et al. (2016) utilized the Sibling Evaluation Questionnaire, UCLA Loneliness Scale, Network Orientation Scale, Family Hardiness Index, and Family Member Well-Being Index to assess various sibling experiences and well-being aspects. Furthermore, Heller et al. (1999) used a questionnaire to evaluate parents’ perceptions of siblings’ interactions with their brothers and sisters with deafblindness ( Table 1 ).

Table 1 . Characteristics of the studies.

Quality of the study

Several limitations and biases have been identified in the examined studies by the authors of each study themselves, highlighting the importance of considering these factors when interpreting the findings. As an example, an interview may lead to the unintended procurement of socially desirable responses, especially when it is based on voluntary participation ( Rowan, 1990 ; Harland and Cuskelly, 2000 ; Laman, 2006 ). Also, as Rowan (1990) mentioned, the understanding of questions by siblings and the author’s interpretation could influence results.

Small sample sizes from specific regions limit the generalizability of the findings ( Rowan, 1990 ; Heller et al., 1999 ; Harland and Cuskelly, 2000 ; Laman, 2006 ; Vert et al., 2016 ). Additionally, three studies did not directly assess the siblings experience but investigated people with deafblindness’ perspective of their siblings’ experience ( Watters et al., 2005 ; Ellis and Hodges, 2013 ) or parents’ perspectives ( Heller et al., 1999 ) ( Table 2 ).

Table 2 . Characteristics of the studies.

Psychological consequences

Siblings may experience certain negative emotions such as resentment towards their parents, primarily due to the parents’ reduced physical and emotional availability ( Watters et al., 2005 ). Some siblings experienced feelings of exclusion and jealousy due to the additional parental attention given to the child with deafblindness ( Watters et al., 2005 ). For example, in the study conducted by Ellis and Hodges (2013) , Bethany (17, type 2) shared her experience of being perceived as the “favorite” among her siblings because she received more attention, which created tension with her sibling. In contrast, another study by Rowan (1990) revealed that nine out of twelve siblings did not perceive their siblings with deafblindness as more fortunate due to the attention or special treatment’ received by the child with deafblindness.

Moreover, in Harland and Cuskelly (2000) study, some siblings expressed guilt over their inability to take a more active role in caring for their disabled sibling. They recognized the burden of care placed on their parents but felt helpless in changing the situation. In this study, one sibling expressed his guilt by saying, “I wish I could do more for Mum because she does do an awful lot.” (p. 300). The same authors noticed that some siblings encounter feelings of anxiety as they grapple with understanding and managing their disabled sibling’s behavior. Interacting with a sibling who has disabilities can indeed present challenges, potentially leading to anxiety and frustration for siblings ( Harland and Cuskelly, 2000 ). Communication barriers, personal insecurities, and the uncertainty surrounding their sibling’s receptiveness to communication may contribute to feelings of exclusion and insecurity ( Harland and Cuskelly, 2000 ).

Also, siblings may experience a sense of inequity and frustration because of the family’s financial problems that are caused by having a child with deafblindness ( Watters et al., 2005 ). Additionally, in Rowan study’s (1990), some siblings mentioned that they felt they bore additional responsibilities which caused frustration and a sense of unfairness. In Rowan study’s, some siblings (3 out of 12) stated they put pressure on themselves to succeed academically or in other areas to compensate for the limitations of their sibling with deafblindness. This self-imposed pressure could contribute to their anxiety. Social embarrassment due to friends’ reactions and questions further exacerbated the emotional toll ( Rowan, 1990 ).

Anxiety may also arise from difficulty envisioning the future. Indeed, sibling disability can significantly impact an individual’s perception of their future, as they envision a lifelong role in the life of the child with deafblindness ( Harland and Cuskelly, 2000 ; Laman, 2006 ). In Laman (2006) study, all the siblings expressed concerns and anxieties regarding their future roles once their parents could no longer support the person with disabilities. Those results are supported by Harland and Cuskelly (2000) results. In Harland and Cuskelly’s study, some siblings also expressed anxiety and concerns for the future. The siblings recognized the importance of future financial support. They acknowledged that their roles would need to evolve as their parents could not provide primary home-based care for their siblings. Furthermore, many siblings expressed inadequacy in not being able to assume greater responsibility for supporting their siblings. This sense of inadequacy was influenced by various factors, resulting in stress and conflict among most siblings ( Harland and Cuskelly, 2000 ). Time constraints emerged as a concern for five out of twelve of the siblings in Rowan (1990) . They expressed worries about having limited time to support their siblings with disabilities due to their numerous other commitments.

Furthermore, some siblings experienced deep distress regarding their lack of preparedness for their future responsibilities towards their sibling with deafblindness. Despite the anxiety stemming from their parents’ mortality, all the siblings were willing to assume increased responsibility if necessary to support their siblings with disabilities ( Harland and Cuskelly, 2000 ). In Harland and Cuskelly (2000) study, most siblings were expected to continue their responsibility for supporting their sibling’s personal development. However, some siblings, like Phillip (a sibling interviewed by Harland and Cuskelly, 2000 ), doubted their ability to take on teaching responsibilities themselves.

The search for suitable accommodation for their sibling with deafblindness was identified as a significant concern for some siblings. In Harland and Cuskelly (2000) study, one sibling stated, “There is nothing for the deafblind in the area of job preparation and placement” (p. 302). Another sibling was concerned about her ability to find appropriate accommodation for her sister in the future. Some siblings also expressed anxiety about the level of care their brother or sister would receive in non-family-based supported accommodation settings ( Harland and Cuskelly, 2000 ). Assisting with or managing their sibling’s financial affairs was another responsibility that most siblings anticipated undertaking in the future ( Harland and Cuskelly, 2000 ). Rowan (1990) stated that the worries and concerns expressed by some siblings of children with deafblindness encompassed fears, concerns, worries and anxieties about their sibling’s mobility, mortality, future employment prospects, and access to education ( Rowan, 1990 ).

Harland and Cuskelly (2000) noticed that some siblings expressed previous apprehensions about the potential of having children with disabilities themselves. One participant in their study sought genetic counseling to address this concern, and another one worried that prospective partners might mistakenly assume genetic complications associated with their relationship.

The experience of having a sibling with deafblindness can give rise to a strong need for information among siblings, leading to confusion, incomprehension, and anxiety. Understanding the specific problems faced by the child with deafblindness is another significant concern for siblings. Some siblings expressed desire to gain deeper insights into terminology, causes of the handicap, functioning abilities, caregiving techniques, preferences, and future prospects for their sibling ( Rowan, 1990 ). They may desire to understand the nature and extent of their sibling’s disability in hopes of finding a sense of clarity about what it means to be deafblind ( Rowan, 1990 ). They questioned whether their sibling is truly deaf, blind, or both, highlighting the need for accurate information to dispel misconceptions ( Rowan, 1990 ). In addition to seeking information, siblings recognized their responsibility as advocates for their siblings with deafblindness. They strongly desired to learn more about available services and opportunities, understanding that this knowledge is crucial for their future role. However, the lack of information leaves them uncertain about their sibling’s future plans and legal matters, causing anxiety and a need for clarity ( Harland and Cuskelly, 2000 ). Concerns about future living arrangements and the potential changes in their sibling’s condition further contribute to siblings’ anxiety. Some siblings believed that their sibling with deafblindness may not always live at home, while others believed their sibling’s deafblindness may change over time ( Harland and Cuskelly, 2000 ). In Heller et al. (1999) study, 67% of the parents recognized the siblings’ desire to learn more about interacting and communicating with their sibling with deafblindness, emphasizing the importance of knowledge and understanding in facilitating meaningful connections.

Banta (1979) highlighted the siblings’ greater distress compared to their parents as a result of ineffective coping mechanisms. As Harland and Cuskelly (2000) noticed, siblings relied heavily on their parents for help and guidance in caring for their sibling with deafblindness. They sought assistance in understanding the special attention and care required by a child with deafblindness, learning how to use specialized equipment, effectively communicate, handle emotional situations, and provide proper nourishment ( Harland and Cuskelly, 2000 ). To cope with their worries and problems, siblings actively sought emotional support from various sources. Mothers are often their primary confidants, followed by fathers and friends ( Rowan, 1990 ) ( Table 3 ).

Table 3 . Results.

Social consequences

Siblings may experience difficulties in interaction with their deafblind brother or sister. Indeed, the presence of communication difficulties related to the nature of the disability, which affects language, makes it difficult for siblings to engage in activities with their sibling with deafblindness ( Heller et al., 1999 ; Watters et al., 2005 ). Communication is an important aspect of the sibling relationship that often needs adjustments and adaptations, commonly achieved through augmentative and alternative methods of communication. In the case of siblings with a brother or sister with deafblindness, it has been observed that they mostly rely on nonsymbolic forms of communication to interact. However, there is a noticeable absence of alternative communication methods, such as tactile or visual communication boards/systems, and limited involvement from professionals in teaching siblings how to communicate with their sibling with deafblindness. Interestingly, most siblings acquired nonsymbolic communication skills independently, as reported by 62.5% of parents and 56.3% of parents of elementary school children ( Heller et al., 1999 ). On the other hand, sign language is predominantly taught by a parent/relative or, in some instances, by a speech therapist, as seen with two older children. Among parents who reported that siblings face communication challenges consistently, those who rely on nonsymbolic forms of communication experienced the greatest difficulty. Additionally, it seems that the nature of these difficulties is influenced by the severity of the impairment and the availability of alternative communication methods ( Heller et al., 1999 ).

In Heller et al. (1999) study, most parents (over 25%) reported that siblings have minimal interaction or deliberately try to avoid their brother or sister with deafblindness. Many parents (22.2%) and parents of elementary school children (16.7%) indicated that siblings never try to engage their brother or sister in any activity. Regarding the duration of sibling engagement, approximately 23.5% of parents and 21.7% of parents of elementary school children reported a daily involvement of 5–15 min, while 20.6% of parents and 26.1% of parents of elementary school children reported spending 30 min to an hour together. However, a notable percentage of parents (14.7%) and parents of elementary school children (21.7%) mentioned that siblings spend less than 5 min with their sibling with deeafblindness, and 17.6 and 8.7% of parents and parents of elementary school children, respectively, stated that no dedicated time is spent between siblings. The frequency of difficulty encountered in playing or engaging in activities together varied among respondents. The majority of parents (44.4%) and parents of elementary school children (41.7%) reported that such difficulties occurred some of the time. When questioned about the siblings’ willingness to make necessary adjustments for their brother or sister with deafblindness to participate in games or activities that require visual perception, a significant percentage of parents (68.6%) and of parents of elementary school children (70.8%) indicated that siblings never made adaptations or only did so on certain occasions. Similarly, for activities requiring hearing, most parents (70.6%) and parents of elementary school children (73.9%) reported that siblings never made accommodations or only did so sometimes.

According to Rowan (1990) , siblings of individuals with deafblindness play pivotal roles in family dynamics, specifically in caring for and supporting their sibling with deafblindness. In Rowan (1990) , a majority (eight out of twelve) indicated that their parents expected them to take on additional responsibilities due to the presence of a sibling with deafblindness. These responsibilities span from babysitting their deafblind sibling and other siblings, assisting younger siblings with chores, bathing, feeding, changing diapers, dressing, providing tracheostomy care, engaging in playtime, attending to the child’s needs during the night, cooking, and undertaking household to outdoor tasks. This underscores the heightened responsibility these siblings’ experience, with two of the twelve respondents citing exclusive household roles arising from their deafblind sibling’s needs ( Rowan, 1990 ). Additionally, six of eleven siblings noted their caregiving role was more extensive than their peers’ involvement with their own siblings. However, ten out of twelve siblings perceive their parents’ rules as fair, indicating a balanced approach to managing family dynamics. Concerning knowledge and skills, six out of twelve siblings reported being adequately equipped to assist their sibling with deafblindness ( Rowan, 1990 ).

In Heller et al. (1999) study, parents characterized the relationship between siblings and the child with deafblindness as “helping.” The study revealed that while mothers were the primary caregivers, siblings played a significant albeit secondary role in supporting their brother or sister with visual and hearing impairments. Harland and Cuskelly (2000) found in their study that most siblings provided practical assistance to their brother or sister with deafblindness (e.g., in mobility, recreational activities, and respite care). In some cases, they even took on the role of being a parent for two weeks to give their parents a break ( Harland and Cuskelly, 2000 ). Most siblings offered practical assistance by reinforcing or teaching their brother or sister with deafblindness new skills. One common responsibility shouldered by adult siblings was maintaining regular contact with their siblings with disabilities. In fact, adult siblings maintained regular contact and emotional support, reinforcing or teaching new skills regardless of geographic proximity ( Harland and Cuskelly, 2000 ).

Siblings of children with deafblindness encounter various challenges and disadvantages in their everyday lives. Public attention when accompanying their sibling with deafblindness and crowded living conditions due to special equipment are among the drawbacks ( Rowan, 1990 ). Frequent hospital visits further add to the siblings’ inconvenience and disruption of their personal routine. Another disadvantage is the impact on their participation in family activities, as their sibling’s condition may require adjustments or prevent them from fully engaging in shared experiences ( Rowan, 1990 ). In Rowan (1990) , nine out of twelve siblings expressed dissatisfaction with how their brother or sister with deafblindness interfered in their lives. This dissatisfaction was often associated with the inability to implement family plans and activities due to their sibling’s needs (i.e., a parent staying home with the child with deafblindness rather than joining the rest of a family on an outing). Furthermore, four out of ten siblings shared their discontent with their sibling’s impact on their personal plans and activities. These concerns range from being unable to visit friends’ houses or host them at their homes, feeling obligated to be involved with the care of their sibling with deafblindness, and having their activities disrupted due to the responsibility of looking after their other siblings. To address the limited participation in activities, it is recommended, within the literature, to modify the activities to accommodate the vision and hearing loss of the child with deafblindness. This modification can enhance siblings’ ability to participate in and foster meaningful interaction. However, it is worth noting that siblings may lack information on how to effectively modify or identify suitable activities for their siblings with deafblindness ( Rowan, 1990 ).

Rowan (1990) also notes the generally positive and protective attitudes of siblings towards their sibling with deafblindness. Notably, in Rowan (1990) all siblings demonstrated a favorable attitude and actively discouraged any teasing directed towards their sibling with deafblindness. Despite the challenges they may encounter, siblings cited genuine enjoyment/happiness stemming from their relationship with their brother/sister with deafblindness and are committed to safeguarding their well-being when necessary. Among the surveyed, six out of the twelve siblings mentioned that their friends visit their home and engage in various activities, such as playing pat-a-cake, throwing a ball, holding hands, and communicating with the child with deafblindness. On the contrary, six siblings reported that their friends do not come to their homes to interact with their siblings with deafblindness. When explaining their sibling’s condition to friends, five out of the twelve siblings expressed no difficulty conveying the necessary information. Feelings of rejection were minimal (eight out of twelve), with most embracing their sibling’s condition openly, showing unconditional love and acceptance. Several stated they did not wish for their deafblind sibling to be absent, highlighting the depth of their emotional bond ( Rowan, 1990 ).

However, Vert et al. (2016) study found a correlation between siblings’ level of difficulty in engaging with their sibling with deafblindness and their willingness to include them in activities with friends or attend support groups. This suggests the potential benefit of interaction with peers who also have siblings with disabilities ( Table 3 ).

While studies have explored the experiences of siblings of individuals with disabilities, the literature on siblings of those with deafblindness remains notably sparse. The existing research does, however, illuminate the unique psychological and social challenges these siblings face.

To summarize, the psychological consequences experienced by siblings of deafblind individuals can include a range of negative emotions such as resentment, jealousy, frustration, guilt, anxiety, and feelings of inadequacy. Siblings may feel resentment towards their parents due to reduced attention and availability, as well as jealousy towards the additional parental attention given to the deafblind child. They may also feel excluded and experience a sense of inequity and frustration. Siblings often express guilt over their inability to take a more active role in caring for their disabled sibling and may experience anxiety when managing their sibling’s challenging behavior. Communication barriers, personal insecurities, and uncertainty about their sibling’s receptiveness to communication can contribute to feelings of exclusion and insecurity. Siblings may also experience anxiety when envisioning the future, particularly regarding their own future roles, once their parents can no longer provide support for the person with deafblindness.

Social consequences for siblings of individuals with deafblindness can include difficulties in interacting with their siblings due to communication challenges related to the disability. Siblings often rely on nonsymbolic forms of communication to interact. Still, there may be a lack of alternative communication methods and limited involvement from professionals in teaching siblings how to communicate with their siblings with deafblindness. Siblings also take on significant responsibilities within the family dynamics, such as caregiving tasks and support for their sibling with deafblindness which impact the time they have for their social activities. In the context of family activities/outings, siblings may experience disadvantages, such as attention and stares from others, routines disruptions, and limited household space. Siblings may also encounter difficulties forming and maintaining relationships outside the family due to their sibling’s condition. Some siblings may actively avoid or have minimal interaction with their deafblind sibling.

Similar experiences have been observed in siblings of children with other types of disabilities. For example, some studies on individuals with undefined physical or chronic disabilities demonstrated that siblings of children with disabilities can experience distress, unhappiness and resentment ( Lamarche, 1985 ; Fisman et al., 2000 ; Scelles, 2011 ; Giallo et al., 2012 ; Hallberg, 2013 ).

Also, siblings of children with other disabilities (undefined physical or intellectual disabilities) can feel ashamed of how others perceive them and struggle with social inclusion ( Lamarche, 1985 ; Dayan and Scelles, 2017 ).

Siblings of children with other disabilities (hemiparesis and undefined physical or intellectual disabilities) may exhibit premature and hyper-protective behaviors, feel responsible for the family’s well-being, and seek partners who understand their role ( Seligman, 1983 ; Hannah and Midlarsky, 1985 ; Scelles, 2004 ; Gardou, 2012 ; Dufreche Rastello, 2020 ).

In addition, siblings of children with other disabilities (undefined chronic illness) tend to have higher anxiety levels stemming from uncertainty in interacting with the disabled sibling ( Heiney et al., 1990 ; Martinez et al., 2022 ).

Finally, siblings of children with hemiparesis express the same lack of information about the disability ( Dufreche Rastello, 2020 ), as for siblings of individuals with deafblindness. Research indicates that siblings of children with hemiparesis desire more information but fear the implications it may have on themselves ( Dufreche Rastello, 2020 ). Limited contact with professionals and guilt often prevents these siblings from asking questions that might equip them with the information they desire, such as questions that address the nature of their siblings disability ( Smith and Perry, 2005 ).

In contrast, some aspects present in the siblings of children with disabilities were not reported in those of children with deafblindness. Negative emotions such as anger are commonly experienced by siblings of children with other disabilities (undefined physical or intellectual disabilities) ( Seligman, 1983 ; Hallberg, 2013 ). These emotions, however, were not reported in siblings of children with deafblindness within our research. Siblings of children with intellectual disabilities may feel threatened by the possibility of death, disability, or contamination ( McHale and Gamble, 1989 ). Also, guilt can inhibit aggressive feelings and lead to depressive symptoms for siblings of children with hemiparesis or physical or mentaldisabilities ( Meynckens-Fourez, 1995 ; Griot et al., 2010 ; Dufreche Rastello, 2020 ). Feelings of loneliness, lack of social support, and withdrawal are also common challenges faced by siblings of children with facial paralysis or other undefined physical disabilities ( Crocker, 1981 ; Pogossian, 2003 ; Guyard, 2012 ). These issues were not identified in our study. Differences in rules and increased sibling rivalry can lead to aggression and behavioral problems for some siblings of children with facial paralysis or other undefined physical disabilities ( Breslau et al., 1981 ; Crocker, 1981 ; Lamarche, 1985 ; Meynckens-Fourez, 1995 ). Siblings of children with hemiparesis may exhibit behavioral issues to gain attention or wish to be disabled themselves ( Dufreche Rastello, 2020 ). However, behavioral problems for siblings of individuals with deafblindness were not identified.

It is unclear if these aspects are absent, not investigated, or not expressed by the siblings in interviews due to modesty or fear. Also, Smith and Perry (2005) noticed that limited contact with professionals and guilt often prevent siblings of children with a disability from asking questions. Additionally, the desire to appear positive to the interviewer may have led to the concealment of information, such as anger, self- or hetero-aggressive behaviors, social difficulties, and feelings of loneliness. It is also possible that these aspects are less prevalent because the research primarily focused on more well-known syndromes such as Usher and CHARGE, for which more information and support groups are available, potentially introducing a representativeness bias.

For example, in Vert et al. (2016) study, siblings of people with deafblindness reported experiencing somewhat lower levels of loneliness, which contradicts earlier research suggesting higher levels of loneliness among siblings of children with intellectual disabilities ( Rossiter and Sharpe, 2001 ). One possible explanation for this discrepancy is that families with a child with CHARGE syndrome have easy access to support systems through the Internet and various networks. While not many participants actually reported that they attended a CHARGE conference or a support group, the availability of these networks may have helped alleviate any potential feelings of loneliness associated with having a sibling with a disability.

Other factors influencing the results including socioeconomic status, past attendance at a sibling support group, parent stress, family time and routines, family problem-solving and communication, and family hardiness. However, these factors were rarely presented in the selected studies, and thus, we could not examine their detailed impact.

The self-esteem of siblings of children with deafblindness was not explored in our study/ Considering the psychological and social challenges siblings of children with deafblindness are facing, it would be interesting to investigate the consequences on their self-esteem. Previous research suggests that self-esteem in siblings of disabled children (with pervasive developmental disorder) is similar to that of other children ( Ferrari et al., 1988 ). Positive effects such as compassion, empathy, and resilience can develop through the sibling bond ( Powell et al., 1985 ; Metzger, 2005 ; Scelles, 2008 ; Griot et al., 2010 ; Von Benedek, 2013 ). According to Mchale and Gamble (1989) siblings of children with autism may also acquire responsibility and develop good self-esteem ( McHale and Gamble, 1989 ). Also, for siblings of children with a pervasive developmental disorder or hemiparesis, social behaviors, skills, and creative abilities can be enhanced ( Ferrari et al., 1988 ; Dufreche Rastello, 2020 ). The lack of results on these variables may be explained by the fact that research initially focused on negative consequences, given the relatively unexplored nature of the field due to the rarity of the disability.

Limitations

Our research is predicated upon the foundation of preceding studies, which are not without their limitations (e.g., sample size, methodology, and generalizability of results). It is recognized that these limitations can limit our own research. One prominent limitation and bias observed in the antecedent studies is the use of the voluntary participation method. This method has the propensity to introduce bias in the resultant data by attracting individuals with positive attitudes or specific interests, thereby potentially impinging upon the representativeness of the sample and the generalizability of the findings.

Sample size is another limitation— posing challenges in extrapolating the findings to encompass the experience of other siblings of children with deafblindness. It is thus imperative to consider this limitation and its consequential impact when interpreting the findings.

Additionally, relying solely on a single interview with each participant is another significant limitation of the antecedent research and our subsequent use/interpretation of these studies. This approach can influence participant responses, engendering socially desirable answers that may deviate from genuine responses. Consequently, the validity of the results and the comprehension of sibling relationships may be compromised.

Cultural differences can substantially influence the results and impede the transferability of studies concerning the impact of disabilities on families across different cultures. We must recognize and account for these cultural disparities when interpreting and applying the findings.

Furthermore, the inability to access texts not available in English and French presents an additional limitation in our research. This constraint may engender incomplete information and potentially result in overlooking pertinent insights.

It is important to note that specific texts could only be partially accessed in certain cases, with only the title or abstract being available. This further curtails our access to comprehensive information and a holistic understanding of those studies. The absence of full-text access may have omitted valuable details and insights.

The limited number of selected studies and the inability to access texts in other languages underscores the necessity for future research endeavors characterized by enhanced methodologies and augmented sample sizes (that ideally encompass greater diversity). Such endeavors will surmount these limitations and engender more robust, dependable, and generalizable results within the domain of deafblindness research.

Recommendations

Practical implications.

In light of the challenges some siblings face, we recommend providing psychotherapeutic spaces 2 to address their needs ( Rowan, 1990 ; Ellis and Hodges, 2013 ). The research we examined underscores, either explicitly or implicitly, the importance of support and care for siblings in (1) understanding the condition of deafblindness better, (2) expressing their emotions, and (3) acquiring information ( Rowan, 1990 ; Ellis and Hodges, 2013 ). However, such therapeutic spaces for siblings remain limited, potentially due to a lack of awareness among parents and institutions ( Plumridge et al., 2011 ).

To meet these needs, we recommend employing the Taylor’s Siblings Questionnaire to identify the concerns of siblings of children with deafblindness and tailor interventions accordingly ( Rowan, 1990 ). Professionals like psychologists and psychometricians can facilitate sibling groups and offer a safe environment for siblings to express themselves and develop a deeper understanding of disability-related issues ( Pitman and Matthe, 2004 ; Dufreche Rastello, 2020 ). Participation in sibling groups has demonstrated numerous benefits, including reduced anxiety, enhanced self-esteem, and improved communication within the family ( Heiney et al., 1990 ; Smith and Perry, 2005 ; Scelles et al., 2007 ; Plumridge et al., 2011 ; Scelles, 2011 ).

Professionals working with families should acknowledge the impact of disability on both siblings and parents, adopting a comprehensive approach to address their needs ( Vert et al., 2016 ). Support programs should actively involve siblings in crucial educational and transitional meetings and provide sibling workshops to equip them with knowledge and skills ( Laman, 2006 ).

Research implications

Future studies should incorporate diverse data sources to ensure reliable data collection ( Vert et al., 2016 ). Further exploration of the role of interveners in supporting siblings and the implementation of larger-scale studies will contribute to a deeper understanding of their needs ( Vert et al., 2016 ).

In the context of deafblindness, long-term studies focusing on interventions targeting psychological well-being, orientation and mobility, independence development, transition to adulthood, and sibling relationships are warranted ( Rowan, 1990 ). Additional research is needed to delve into the positive aspects, coping resources, and resilience developed by siblings ( Rowan, 1990 ).

To enhance the generalizability of research, it is recommended to include families from diverse geographical regions and incorporate the perspectives of siblings and children with deafblindness ( Rowan, 1990 ). Conducting longitudinal studies on sibling relationships over time would provide valuable insights, particularly for children with deafblindness. This study highlights the importance of addressing limited interaction or avoidance between siblings and their brother/sister with deafblindness by providing tailored information and support for sibling relationships ( Rowan, 1990 ).

Furthermore, future research should focus on larger and more diverse samples, employ improved methodologies, and directly incorporate siblings’ perspectives to ensure more reliable and generalizable results in the field of deafblindness research.

Author contributions

MA did the mini-review. RP leads the research project of which this mini-review is a contribution and coordinated MA’s work during the selection process and participated in the corrections. ND corrected the text and provided perspectives. All authors contributed to the article and approved the submitted version.

Conflict of interest

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

Publisher’s note

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

1. ^ In this context, siblings refer to each of the two or more children or offspring who share one or both parents, specifically brothers or sisters.

2. ^ A therapeutic space is a specially designed environment that supports healing and therapy. It can be a physical or virtual space where individuals engage in therapeutic activities to improve their well-being. It promotes safety, comfort, and relaxation, and may include tools and materials to enhance the therapeutic experience.

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Keywords: deafblindness, siblings, family, psychological consequences, social experiences, siblings’ psychotherapeutic interventions

Citation: Arcous M, Potier R and Dumet N (2024) Psychological and social consequences of deafblindness for siblings: a systematic literature review. Front. Psychol . 15:1102206. doi: 10.3389/fpsyg.2024.1102206

Received: 18 November 2022; Accepted: 04 March 2024; Published: 25 April 2024.

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*Correspondence: Marine Arcous, [email protected]

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Carbon Offsets and Regulation: A Literature Review

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Carbon offset markets have become increasingly popular avenues for stakeholders, ranging from industries to governments, to invest in emissions reduction projects in developing nations rather than focusing solely on mitigating their own emissions. Originating from the Clean Development Mechanism (CDM) under the Kyoto Protocol, these markets aimed to supplement efforts to meet emission reduction targets. However, the emergence of voluntary carbon mechanisms (VCMs) alongside regulatory CDMs has sparked concerns regarding trust and legitimacy within the carbon offsetting landscape. Issues such as double accounting, economic prioritization over environmental impact, and lack of consistent regulation have raised doubts about the efficacy and fairness of carbon offset markets. This paper examines the causes and consequences of these challenges and proposes potential solutions to address them. It argues for the adoption of standardized regulations, certification processes, and governance structures across both CDM and VCM markets, emphasizing the need for transparency and accountability. Additionally, it explores alternative approaches such as implementing carbon taxes and prioritizing projects focused on conservation and newer technologies. By establishing clear guidelines and fostering collaboration between regulatory and voluntary markets, stakeholders can work towards restoring public trust and ensuring the effectiveness of carbon offset initiatives in combating climate change. 

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Merkel cell carcinoma overlapping Bowen’s disease: two cases report and literature review

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• Published: 26 April 2024
• Volume 150 , article number  217 , ( 2024 )

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• Xueqin Chen 1 ,
• Xiao Song 1 ,
• Hui Huang 1 ,
• Lian Zhang 1 ,
• Zhiqiang Song 1 ,
• Xichuan Yang 1 ,
• Shanchuan Lei 2 &
• Zhifang Zhai 1  

Merkel cell carcinoma (MCC) is a rare neuroendocrine tumor of the skin, which mainly occurs in the sun exposed sites of white patients over 65 years, with a higher recurrence and metastasis rate. Clinically, MCC overlapping Bowen’s disease (BD) is a very rare subtype of MCC. Few cases in the literature have been described and the management is not well defined. We summarize and update the epidemiology, clinical and histopathological features, metastasis characteristics, local recurrence rate and management of it by presenting two cases of MCC overlapping BD and reviewing the literature over the last 11 years.

We consulted databases from PubMed, ResearchGate and Google Scholar by MeSh “Merkel cell carcinoma” and “Bowen’s disease”, “Bowen disease” or “squamous cell carcinoma in situ”, from January 2013 to December 2023 and reviewed the literatures. We reported two additional cases.

Total 13 cases of MCC overlapping BD were retrospectively analyzed, in whom mainly in elderly women over 70 years, the skin lesions were primarily located on the faces, followed by the extremities and trunk. Most of them were asymptomatic, firm, dark red nodules arising on rapidly growing red or dark brown patches, or presenting as isolated nodules. Dermoscopy evaluation was rarely performed in the pre-operative diagnostic setting. All cases were confirmed by histopathology and immunohistochemistry. The most definitive treatment was extended local excision, but local recurrences were common. Of the 13 cases, 4 cases experienced local or distant metastasis. One suffered from an in-transit recurrence of MCC on the ipsilateral leg after local excision and lymph node dissection, whose metastasis completely subsided after avelumab treatment and without recurrence or metastasis during 6 months of follow-up.

Conclusions

MCC overlapping BD is a very rare skin tumor mainly predisposed on the faces, with high misdiagnosis rate and recurrence rate. Advanced disease at diagnosis is a poor prognostic factor, suggesting that earlier detection may improve outcome. The acronym, AEIOUN, has been proposed to aid in clinical identification. Our reports and the literature review can provide a better awareness and management of it.

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Introduction

Merkel cell carcinoma (MCC) is a rare and highly aggressive primary cutaneous neuroendocrine carcinoma, which predominantly affects individuals of Caucasian descent. Risk factors include advanced age, exposure to ultraviolet radiation, male gender, immunosuppression, hematologic malignancies or posttransplant status, and infection with Merkel cell polyomavirus (MCPyV) (Harms et al. 2018 ). It is characterized by high invasiveness, frequent local recurrence, a tendency for regional lymph node and distant metastases, with high mortality rates of 33–46% (Harms 2017 ; Garcia-Carbonero et al. 2019 ).

MCC often occurs in sunexposed sites, typically presenting as solitary nodules or patches with skin-colored, red or purple hues. Reportedly, it occured concomitantly with or in the setting of pre-existing cutaneous neoplasms, including actinic keratosis, Bowen’s disease (BD), squamous cell carcinoma (SCC), basal cell carcinoma (BCC) and miscellaneous adnexal tumors (Kervarrec et al. 2022 ). Only a small percentage of MCC presented combined with other tumors, for which, current data have suggested a more aggressive course than pure MCC (Tono et al. 2015 ; Chattopadhyay et al. 2020 ). Clinically, its association with BD is exceedingly uncommon. Its unspecific manifestations often lead to delayed diagnosis clinically, which is necessary for dermatologists and oncologists to familiarize themselves with and recognize it (Swain et al. 2022 ).

Few cases in the literature have been described and the management is not well defined. In our paper, we reviewed the literature and reported two additional cases to summarize the epidemiology, clinical and histopathological characteristics and management of it.

We first reported two cases with MCC overlapping BD. Then, we searched different databases, including PubMed, ResearchGate and Google Scholar by the combination MeSh of “Merkel cell carcinoma” and “Bowen’s disease, “Bowen disease” or “squamous cell carcinoma in situ” from January 2013 to December 2023. Total 15 papers were identified. Inclusion criteria were systematic review or meta-analysis of randomized controlled trials, review, retrospective comparative reviews/studies and case series. Exclusion criteria were laboratory studies and non-English translated articles. A wide review of the bibliography of each of the selected articles was performed. In total, 10 papers met our inclusion criteria, including 11 case reports and case series.

We reviewed and analyzed all of the cases with MCC overlapping BD and summarized the demographic information, such as the age, sex and the medical history, clinical and histopathological characteristics and the treatment and prognosis of them.

Case reports

Case 1 A 51-year-old man presented with a pruritic erythema on the right waist for over 5 years. In the past years, he paid no attention to it, though a dull red nodule had developed and gradually enlarged on the erythema. Physical examination revealed a 5 cm × 3 cm oval-invasive erythema covering with some scale, in the center of which a dull red, solid, non-tendor, well-demarcated nodule measuring 3 cm × 2 cm × 2 cm protruded from the skin surface. Significant hyperplasia of dilated capillaries, and a few scales can be seen on the surface of the neoplasm (Fig. 1 a). Thoracoabdominal CT revealed no significant abnormalities, and peripheral blood count and tumor marker tests were normal.

Case 2 An 87-year-old female presented with an asymptomatic neoplasm on the right maindibular angle for over a year. Physical examination showed a dull red patch measuring 2 cm × 1.5 cm , with a solid, well-defined and protruding nodule in the center measuring  1.5 cm × 1.5 cm × 1 cm, and with some scaling (Fig. 1 b). There were no positive findings by the cranial and thoracoabdominal CT scans.

Biopsies were performed respectively on the neoplasms in both cases. Both the histopathological examination of the two cases revealed gross hyperkeratosis with parakeratosis overlying a thickened dysplastic epidermis, with the atypical mitoses and multinucleated tumor giant cells. A small blue cell tumor extended deeply into the subcutaneous fat under the low-power magnification, and the pathognomonic tumor nuclei were large and pale staining and contain tiny nucleoli (Fig. 2 a–f). Immunohistochemistry of case one showed positivity for CK, CK20, EMA, Synaptophysin (Syn) and Bcl-2 (Fig. 3 a–d). Vimentin was positive in the stroma, while CD3, CD4, CD8 and CD20 showed scattered positivity. Ki67 was positive in 90% of tumor cells. LCA, CD68, CD30, TdT, CD56, Mum-1, TIA1, Granzyme B, EBER, Neuron-Specific Enolase (NSE), Chromogranin A (CgA) and CD79a were negative. In case two, immunohistochemistry showed positivity for CD56, focal positivity for CK, CK20, CAM5.2 and CgA, and negativity for Syn and CK7. Ki67 was positive in 80% of cells (Fig. 4 a–f)

Clinical aspect of an MCC overlapping BD. a Solitary and dome shaped reddish nodule surrounded by an erythematous scaly patch on the right waist. b Ovoid dark erythematous painless tumor mass on the right mandibular angle, with peeling and scabbing on the surface of the mass

a, b On histopathology, BD is juxtaposed or strictly intermingled with MCC (H&E, ×4). c, d BD shows full thickness of atypical squamous cells (H&E, ×10). e, f Dermal dense infiltration of small round hyperchromatic small cells (H&E, ×10)

a–c Immunohistochemical staining showed that CK, CK20 and Syn were positive Magnification: ×10. d Immunohistochemical staining showed that Ki67 was positive in 90% of the cells Magnification: ×10

a Immunohistochemical staining showed CD56 positive Magnification: ×10. b–e Immunohistochemical staining showed that CK, CK20, CAM5.2 and CgA were focally positive Magnification: ×10. f Immunohistochemical staining showed that Ki67 was positive in 80% of the cells Magnification: ×10

Both patients were diagnosed with MCC overlapping BD. They all underwent surgical excision extending 1 cm beyond the tumor margins. There was no recurrence during a follow-up period of 3 years in case one and about half a year in case two.

Literature review

The demographic data.

In the last 11 years (from 2013 to 2023), only 13 cases (including our two cases) of MCC overlapping BD have been described in the literature. The incidence was slightly higher in females than in males, with a male-to-female ratio of 1:1.6 (5 cases to 8 cases). The age ranged from 32 to 87 years (mean 72 years, median 73 years). Lesions mainly occurred at the age of more than 70 years (77%), and only one extremely rare case occurred at the age of 32 years. The overall duration of the disease varied from 2 months to 5 years. Some patients had a history of annual herbal pill consumption, exposure to ultraviolet radiation, and previous diagnoses of multiple myeloma, basal cell carcinoma, and BD (Choe et al. 2014 ; Miraflor et al. 2016 ) (Table 1 ).

Clinical manifestations

All the studies reported the location of the lesions. MCC overlapping BD were mostly located on the faces ( N = 7/13, 53.8%), followed by the trunk ( N = 3/13,23.1%), the upper extremity ( N = 1/ 13,7.7%) , lower extremity ( N = 1/ 13,7.7%) and groin ( N = 1/ 13,7.7%). No patient had multiple lesions (Table 1 ).

Information regarding the initial clinical presentation was available for all patients. The lesions were most frequently described as asymptomatic, firm, dull red nodules on red or dark brown patches with frequent rapidly growing behavior, or as solitary nodules. None of the lesions described with accuracy were correctly diagnosed before biopsy and histological examination. The size of the tumor lesions was available for 11 lesions (84.6%). Tumor diameters ranged from 0.3 to 6.5 cm (mean: 2 cm, median: 1 cm). Rapid growth, either of new lesion or stable lesion from several months was the most frequent motivation for biopsy and diagnosis (Table 1 ).

Locoregional or distant metastases occurred in four patients (30.8%) (Swain et al. 2022 ; Choe et al. 2014 ; Ishida et al. 2013 ; Kiyohara et al. 2019 ). One patient showed lymph node and liver metastasis (Choe et al. 2014 ). After local excision of the cutaneous lesion and left inguinal lymph node dissection in one patient, several dermal and subcutaneous nodules developed successively on the left lower extremity (Kiyohara et al. 2019 ) (Table 1 ).

All 13cases had a histological diagnosis of MCC overlapping BD (Table 1 ). Eight patients were diagnosed by histopathology, followed by extensive local excision treatment. Of them, an 82-year-old female was diagnosed with MCC overlapping BD by histopathology but refused further evaluation and operative treatment (Jeong et al. 2018 ). Seven patients underwent direct extensive local excision treatment, followed by histopathological detection of the tissue post-surgery (Tono et al. 2015 ; Swain et al. 2022 ; Choe et al. 2014 ; Miraflor et al. 2016 ; Ishida et al. 2013 ; Kiyohara et al. 2019 ; Yamamoto 2014 ; McGowan et al. 2016 ; Casari et al. 2018 ). A 77-year-old woman was found to have lymph node and liver metastases after surgical treatment, followed by radiation and chemotherapy (Choe et al. 2014 ). A 65-year-old Japanese man experienced recurrent skin lesions after local surgery and lymph node clearance. After receiving avelumab treatment for 2 months, all lesions disappeared completely. Subsequent follow-ups over six months showed no recurrence (Kiyohara et al. 2019 ). A 32-year-old lady underwent surgery, lymph node clearance and received radiation therapy. This patient had an axillary dissection because of a palpable lymph node. Two lymph nodes out of 14 showed metastatic deposits, hence the female patient received radiotherapy after which she is well and completely free of disease now, 7 years after the initial diagnosis (Swain et al. 2022 ). A 71-year-old Caucasian male remained recurrence-free during the 1-year follow-up after surgical treatment (Miraflor et al. 2016 ). The follow-up status for the remaining seven patients has not been reported.

• Histopathology

Histopathologically MCC primarily locates within the dermis and can invade subcutaneous tissues. At low magnification, it appeared as a typical small round blue-cell tumor, comprising three different histologic subtypes: trabecular type, intermediate type and small cell type. Among them, the intermediate type was the most common. The tumor consisted of nodules and diffuse sheets of basophilic tumor cells with vacuolated, pale-staining nuclei containing small nucleoli. The cytoplasm was indistinct with common nuclear folding. The trabecular type, the least common, was composed of slender, uniformly shaped cells, often with nuclear folding. The small cells type was characterized by infiltrates of deeply staining 'oat cell-like' cells with significant cell fragmentation.

The histopathological feature of MCC overlapping BD include abnormal keratin-forming cells of BD within the epidermis and small round blue-staining cells of MCC in the dermis. There has been a case reported where the MCC component, in association with BD, was confined to the epidermis, referred to as “intraepidermal MCC”(Miraflor et al. 2016 ).

The immunohistochemical characteristics of MCC overlapping BD indicate that MCC cells express neuroendocrine markers such as NSE, CK20, Neurofilament (NF), CgA, and Syn. Most MCCs do not express Thyroid Transcription Factor-1 (TTF-1). On the other hand, BD commonly exhibits expression of squamous cell markers like CK5/6, CK10 and CK14.

Dermoscopic examination was only reported in two patients (Casari et al. 2018 ). Dermoscopic examination showed the presence of clustered dotted vessels over a reddish structureless area that was suggestive for the diagnosis of BD. Addittional dermoscopical characteristic of the nodule included an atypical vascular pattern with tortuous vessels overlying a whitish background.

MCC is a primary cutaneous neuroendocrine carcinoma, predominantly diagnosed in fair-skinned elderly populations. Characterized by its aggressive nature, MCC is particularly notorious for its tendency towards local recurrences and distant metastases (Siqueira et al. 2023 ). Interestingly, MCC lesions may coexist with, or be found in close proximity to, a variety of other neoplasms, including actinic keratosis, BD, invasive SCC, BCC and sweat gland tumors (Kervarrec et al. 2022 ; Hobbs et al. 2020 ). Clinically, cases of MCC coexisting with BD are very rare. This comprehensive review of the literature spanning the past decade has revealed a notably rare occurrence, identifying only 11 cases of MCC overlapping BD. At the same time, we reported another two cases with MCC overlapping BD in the paper.

The annual incidence rate of MCC is approximately 0.24 cases per 100,000 individuals, showing a trend of exponential increase. This rising incidence can be attributed to a confluence of factors, including demographic shifts towards an aging population, heightened use of immunosuppressive agents, significant improvements in diagnostic technologies facilitating earlier and more accurate detection, and a general enhancement in clinical vigilance and awareness regarding this malignancy (Mistry et al. 2023 ; Mohsen et al. 2023 ). Our retrospective examination has disclosed that incidences of MCC overlapping BD present a gender distribution, with a male-to-female ratio of 1:1.6. Notably, a higher prevalence is observed in females, a finding potentially attributable to the limited scope of the sample size. The pathological manifestations of MCC predominantly arise in individuals aged over 70, though a sporadic occurrence in middle-aged adults has been noted (Swain et al. 2022 ). Owing to the absence of a conclusive diagnosis in MCC patients before undergoing histopathological examination, recent research has offered valuable insights into the clinical presentation of MCC, coalescing into the AEIOU mnemonic for ease of recall. “A” stands for asymptomatic lesions, often presenting without pain or tenderness. “E” denotes rapid expansion, with lesions demonstrating notable enlargement over a period of just three months. “I” represents immunosuppression, a key risk factor, encompassing conditions such as HIV infection, post-solid organ transplantation, or chronic lymphocytic leukemia. “O” refers to individuals over the age of 50, a demographic showing increased susceptibility. Finally, “U” highlights ultraviolet exposure in fair-skinned individuals as a significant risk factor. In the context of MCC overlapping BD, we propose the “AEIOUN” guideline, wherein “N” signifies the rapid emergence of nodules on the foundation of erythematous patches, expanding upon the initial AEIOU criteria.The presence of three or more of these features warrants a heightened clinical suspicion of MCC or MCC overlapping BD, guiding the clinician towards appropriate diagnostic and therapeutic interventions (Siqueira et al. 2023 ; Brusasco et al. 2022 ; Mistry et al. 2023 ).

The etiology of MCC is multifactorial. Current research posits that infection with MCPyV and genetic mutations triggered by ultraviolet (UV) radiation are primary contributors to the pathogenesis of MCC (Yang et al. 2022 ). Specifically, UV radiation plays a critical role in the development of MCPyV-negative MCC cases. Intriguingly, MCC cases that present concurrently with SCC or BD predominantly lack MCPyV. Among the 13 cases of MCC overlapping BD that were reviewed, only three patients underwent testing for MCPyV, and all 3 cases were negative for MCPyV. This observation suggests a distinct oncogenic mechanism in composite MCC, diverging from the pathways observed in solitary MCC (Kervarrec et al. 2022 ). The simultaneous manifestation of MCC overlapping BD might be attributed to a confluence of various factors, including immune regulation and the intricacies of the tumor microenvironment (Chattopadhyay et al. 2020 ). Genetic predisposition also appears to play a significant role in this context. Moreover, the pathogenesis of these conditions may be exacerbated by immunosuppression, immune deficiency or immune system dysregulation. The interplay and communication among cells within the tumor microenvironment, particularly through the release of cytokines, are believed to significantly influence the coexistence of MCC overlapping BD. Furthermore, lifestyle choices and environmental exposures, such as to chemicals, ultraviolet radiation, or toxins, are potential contributory elements in the concurrent development of these dermatological conditions (Casari et al. 2018 ).

MCC is unequivocally diagnosed through histopathological examination, recognized as the definitive gold standard. This is often supplemented by immunohistochemical profiling to accurately distinguish MCC from other poorly differentiated neoplasms. A significant majority of MCC cases demonstrate cytokeratin expression, with approximately 95% exhibiting perinuclear and/or cytoplasmic positivity for CK20 or CAM5.2. Additionally, these carcinomas frequently express neuroendocrine markers, most notably Syn, CgA, CD56 and NF. In contrast, TTF-1 and CDX-2 are typically negative in MCC (Khanna et al. 2020 ). Emerging studies suggest that around 60% of MCC cases express the protein p63, which is potentially correlating with a decreased overall survival rate and a lower disease-specific survival rate of patients. CK7 expression is generally absent in MCC, while there are noteworthy instances of CK7 positivity. A notable aspect in the immunohistochemical landscape of MCC is positive for MCV, observed in about 55% to 90% of cases. Interestingly, MCV-negative cases frequently demonstrated a lack of NF expression, and they distinctively exhibit markers of follicular stem cells along with a higher incidence of p53 positivity, which are predominant in CK20-negative MCCs. In patients with MCC overlapping BD, a distinctive dual pathology is often revealed through marker expression. The immunohistochemical signature of MCC is characterized by the concurrent expression of epithelial markers, including AE/1AE3, CAM5.2 and a broad spectrum of cytokeratins, alongside neuroendocrine markers such as neurofilaments and neuron-specific enolase. In contrast, BD typically demonstrates positive immunoreactivity for markers like CK7 and p16. Notably, an uncommon immunophenotype has been documented in a case merging MCC overlapping BD. In some cases, the MCC cells exhibited an absence of CK20 expression while maintaining positivity for CK7.

This diagnostic approach assumes critical importance in instances where MCC coexists with other tumors derived from epidermal origins. In conducting histological evaluations, it is imperative to scrutinize for the co-occurrence of other tumor types, such as SCC (observed in up to 15% of cases) or BCC. A critical aspect of the assessment is determining the extent of epithelial involvement by the tumor, whether the tumor cells are situated within the epidermis or affect the cutaneous adnexa. Furthermore, a detailed examination of the tumor's morphological attributes is essential, including discerning whether the tumor presents as infiltrative or manifests as well-defined nodular formations, alongside evaluating the dimensions (ranging from small to large) and the particular morphology of the cells, which can determine the appropriate treatment strategies and the prognosis of the tumors. The extent of invasion, growth patterns, and overall prognosis of MCC tumors exhibit significant interrelations (Gonzalez et al. 2022 ). Notably, patients exhibiting sentinel lymph node involvement typically will face a more challenging prognosis. Similarly, individuals suffering from active hematologic malignancies or under immunosuppression are also likely to experience adverse outcomes. Research on the differential prognosis or metastasis rates between MCC and MCC overlapping BD remains scarce. However, the co-occurrence of MCC overlapping BD may signal a broader spectrum of skin damage and an elevated risk of recurrence or metastasis.

The treatment of MCC overlapping BD adheres to the principles established for MCC treatment (Green et al. 2022 ; Harvey et al. 2022 ). In the management of MCC, surgical excision is often considered the primary modality of treatment. This involves employing techniques such as Mohs micrographic surgery or its modified forms, ensuring a margin of 1–2 cm extending to the fascia or periosteum (Uitentuis et al. 2022 ). For metastatic, post-surgical residual, or recurrent MCC, a combination of radiotherapy and chemotherapy serves as effective adjunctive treatment modalities. In recent advancements, immunotherapies targeting various anti-tumor immune mechanisms, particularly therapies focused on the PD-1 and PD-L1 pathways, have emerged as frontline treatments for metastatic MCC (Harms et al. 2018 ; Becker et al. 2018 ). Agents such as Avelumab, Pembrolizumab, and Nivolumab have been instrumental in significantly prolonging patient survival in these cases (Fojnica et al. 2023 ; Topalian et al. 2020 ).

The coexistence of MCC overlapping BD represents an exceedingly rare condition, necessitating further research and accumulation of cases to better comprehend its clinical characteristics and determine the optimal therapeutic regimen. It is a lesion with nonspecific features and dermoscopy evaluation can be helpful for improving the clinical suspicion. We introduce the concept of “AEIOUN” as a pioneering approach for the early identification of clinically suspicious lesions indicative of MCC overlapping BD. The excision of doubtful nodular lesions is mandatory especially in the elderly, because MCC overlapping BD has not only a tendency to recur locally, but it can also metastasize. However, due to its rarity, there are no well-defined guidelines for the management. Complete surgical excision with clear margins stands as the optimal therapeutic choice, complemented by adjuvant radiotherapy, chemotherapy, immunotherapy, or their combination. Regular follow-ups are strongly recommended to monitor the condition. These two case reports and the review of the literature can provide better awareness and management of this rare tumor.

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Xueqin Chen, Xiao Song, Hui Huang, Lian Zhang, Zhiqiang Song, Xichuan Yang & Zhifang Zhai

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Chen, X., Song, X., Huang, H. et al. Merkel cell carcinoma overlapping Bowen’s disease: two cases report and literature review. J Cancer Res Clin Oncol 150 , 217 (2024). https://doi.org/10.1007/s00432-024-05743-0

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Idiopathic intracranial hypertension associated with SARS-CoV-2 infection in an adult male patient: a case report and review of the literature

• Gashaw Solela   ORCID: orcid.org/0000-0002-2233-9270 1 ,
• Addis A. Tenaw 1 ,
• Henok Fisseha 2 ,
• Abel M. Argaw 2 ,
• Tamirat Petros 2 &
• Betelhem Mengistu 3  

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Headache is a frequent symptom in coronavirus disease 2019 (COVID-19) patients, and idiopathic intracranial hypertension (pseudotumor cerebri) has been reported among patients who underwent lumbar puncture for persistent headaches.

Case presentation

A 45-year-old black man presented with dyspnea, cough, fever and headache for 05 days followed by blurring of vision associated with worsening of the headache. Physical examination was significant for tachypnea and oxygen desaturation and there were no abnormal neurologic findings. He tested positive for SARS-CoV-2 with nasopharyngeal swab PCR. His CSF opening pressure appeared high with normal CSF analysis and brain magnetic resonance imaging (MRI) revealed prominent subarachnoid space around the optic nerves and bilateral papilledema. He had significant improvement with medical therapy alone.

Idiopathic intracranial hypertension (IIH) may occur in association with SARS-CoV-2 infection and should be considered when making a differential diagnosis for headache and blurring of vision. COVID-19 may play a role in the development of intracranial hypertension, even in the absence of known risk factors. Early diagnosis and treatment of IIH has paramount importance to prevent vision loss and other morbidities.

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Coronavirus disease 2019 (COVID-19), caused by severe acute respiratory syndrome coronavirus type 2 (SARS-CoV-2), has been the main global public health concern since March 2020 [ 1 ]. Although respiratory conditions are the predominating manifestations of COVID-19, neurological disorders are also becoming more frequently reported. Well known neurologic disorders reported in COVID-19 include encephalitis, encephalopathy, cerebrovascular illness, and Guillain-Barré syndrome ( 2 ).

Patients with COVID-19 frequently complain of headaches, and idiopathic intracranial hypertension (IIH), also called as pseudotumor cerebri has been observed in those who had lumbar puncture for persistent headaches ( 3 ). Patients with various neurologic conditions associated with COVID-19 have also been reported to have elevated intracranial pressure, usually in a mild form ( 4 ).

Cases reports of IIH associated with COVID-19 have been rare and they are described mainly in children and limited number of adult female patients. To the best of our knowledge, this is the first adult male patient who developed idiopathic intracranial hypertension associated with COVID-19.

A 45-year-old black Ethiopian man presented to our COVID-19 isolation center with intermittent dry cough and dyspnea for 05 days associated with new onset mild holocranial headache, low grade fever, myalgia and arthralgia. One day after his admission to the center, he started to develop blurring of vision associated with worsening of the headache. He also had one episode of projectile vomiting of ingested matter. He had no known chronic medical illnesses and no history of drug intake including vitamin A derivatives and tetracycline before the onset of the aforementioned symptoms.

Physical examination revealed blood pressure of 127/91 mmHg, pulse rate of 98 beats per minute, respiratory rate of 28 breaths per minute, oxygen saturation of 86% without oxygen and 92% with 2 L/min intranasal oxygen and body mass index (BMI) of 22.8 kg/m 2 . He had bilateral coarse crepitation over his lower lung fields. He was conscious with Glasgow Coma Scale (GCS) of 15/15 and all the cranial nerves were intact with normal visual acuity (20/20) and visual fields. Meningeal signs were negative and there were no sensory or motor deficits.

Upon investigations, he tested positive for SARS-CoV-2 with nasopharyngeal swab polymerase chain reaction (PCR) and he had mild leukocytosis with left shift and lymphopenia. Lumbar puncture was performed after doing brain magnetic resonance imaging (MRI) and cerebrospinal fluid (CSF) opening pressure appeared high, though it was not measured. The other laboratory results were non-remarkable (Table  1 ).

Brain MRI revealed prominent subarachnoid space around the optic nerves and bilateral papilledema, but didn’t show any mass lesion, hemorrhage, or cerebrovascular lesion and brain magnetic resonance venography (MRV) was normal (Fig.  1 A and B). Chest x-ray revealed bilateral ground glass opacities mainly in the middle and lower lung zones with right side blunted costophrenic angle which was suggestive of COVID-19 pneumonia (Fig.  1 C).

T2 axial and sagittal brain MRI scan showed prominent subarachnoid space around the optic nerves ( A ) and flattening of pituitary gland ( B ). Chest X-ray showed bilateral ground glass opacities mainly in the middle and lower lung zones with right side blunted costophrenic angle ( C )

The patient initially received supportive therapies for severe COVID-19 infection including IV antibiotics, dexamethasone 6 mg IV daily, intranasal oxygen, and prophylactic dose of unfractionated heparin. Acetazolamide 250 mg three times daily was added after establishing the diagnosis of idiopathic intracranial hypertension. He was then followed clinically if he could have an indication for ventriculoperitoneal shunt but his headache and blurring of vision improved and he did not require any surgical intervention. He was finally discharged after 10 days of inpatient supportive medical treatment with significant improvement except occasional cough and mild headache. He did not have any complaint upon reevaluation on the second week of discharge and the acetazolamide was discontinued. He was then doing well throughout his follow up over 6 months in the outpatient department.

Coronavirus disease 2019 (COVID-19, caused by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), is clinically characterized by fever, myalgia, diarrhea, and respiratory illness ( 5 ). However, a number of neurological manifestations have been linked to COVID-19 in the literature, which can be divided into peripheral nervous system (PNS) manifestations like hyposmia/anosmia, hypogeusia/ageusia, myalgia, and Guillain–Barre syndrome and central nervous system (CNS) manifestations like headache, dizziness, impaired consciousness, acute cerebrovascular disease, and seizure disorders ( 6 , 7 ).

Only a small number of cases of idiopathic intracranial hypertension (IIH) linked to COVID-19 have been reported, the majority of which affected children ( 8 , 9 , 10 ). In one study made on 58 patients with distinct neurological conditions associated with COVID-19, cerebrospinal fluid (CSF) analysis revealed that CSF pressure was elevated in one-third of COVID-19 patients with different neurological conditions and all of these patients presented early with intracranial hypertension and none of them developed loss of vision ( 11 ). In line with the results of this study, our patient appeared to have high CSF opening pressure, though not measured; indicating high intracranial hypertension and he had blurring of vision which subsequently improved with acetazolamide.

Idiopathic intracranial hypertension (IIH) is still exceedingly uncommon with 12 to 20 cases per 100,000 persons per year and the main risk factors are obesity, female gender, and reproductive age ( 13 ). The idea that obesity is an inflammatory illness is growing and markers of inflammation were found in the CSF of individuals with idiopathic IHT ( 13 ). A case series of eight adult patients described an association between IIH and COVID-19 ( 14 ). All of these patients were women; many of them were obese; and most of them improved with medical therapy alone (Table  2 ). Unlike the patients described in this case series, our patient was male and non-obese, but similar to most of these cases, he improved with medical therapy alone.

The cause of IIH in COVID-19 is uncertain and debatable. According to one study, increased CSF pressure is linked to high levels of neurofilament light chain proteins, which may be an indication of an active and exaggerated inflammatory process ( 11 ). Cerebral venous sinus thrombosis (CVT), which has been observed in COVID-19 patients, is another mechanism linked to IIH ( 12 ).

There were two case reports of middle aged female patients who had idiopathic intracranial hypertension and visual loss associated with COVID-19. The first one was a 40-year-old obese female patient, who presented with headache, bilateral optic disc edema, and visual loss, which occurred two weeks after making the diagnosis of COVID-19. Her CSF opening pressure was 410 mmH 2 O, and cranial imaging was normal. Visual loss improved after initiation of accetazolamide 250 mg three times daily ( 15 ). The second case was a 49-year-old non-obese woman, who presented with headache and vision loss and found to have COVID-19. Upon further work ups, she was diagnosed to have COVID-19 related IIH, after which she treated with mannitol infusion and oral acetazolamide 250 mg twice daily and then the headache and visual loss got improved. However, she presented again with acute vision loss, which was managed by endoscopic optic nerve fenestration surgery and then she had a good recovery ( 16 ). Our patient had similar clinical manifestations with the aforementioned cases, though he was male by gender and he did not have severe degree of visual impairment. Besides, he had remarkable improvement with medical treatment (acetazolamide) alone like that of the first case ( 16 ).

Our patient was male and had none of the aforementioned risk factors for IIH like obesity and CVT implying that SARS-CoV-2 infection could be the sole culprit in the development of IIH. To the best of our knowledge, all of the cases reported to have IIH associated with COVID-19 were women and most of them were obese and we reported the first male patient with a case of idiopathic intracranial hypertension (IIH) following COVID-19 infection.

Idiopathic intracranial hypertension (IIH) may occur in association with SARS-CoV-2 infection and should be kept in mind when making a differential diagnosis for headache and blurring of vision. COVID-19 may play a role in the development of intracranial hypertension, even in the absence of known risk factors. Early diagnosis and treatment of IIH has paramount importance to prevent vision loss and other morbidities.

Data availability statement

Data supporting this case report will be available with the corresponding author upon reasonable request.

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Acknowledgements

We would like to thank the patient for approval of publication of his case details.

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Department of Internal Medicine, Yekatit 12 Hospital Medical College, Addis Ababa, Ethiopia

Gashaw Solela & Addis A. Tenaw

Department of Internal Medicine, St. Paul’s Hospital Millennium Medical College, Addis Ababa, Ethiopia

Henok Fisseha, Abel M. Argaw & Tamirat Petros

Addis Ababa City Administration Health Bureau, Addis Ababa, Ethiopia

Betelhem Mengistu

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GS was involved in data curation, validation, and writing–review and editing. AA was involved in conceptualization, data curation and writing–original draft. HF, AM, TP and BM were involved in data curation and writing–original draft. All authors read and approved the final manuscript.

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Correspondence to Gashaw Solela .

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Solela, G., Tenaw, A.A., Fisseha, H. et al. Idiopathic intracranial hypertension associated with SARS-CoV-2 infection in an adult male patient: a case report and review of the literature. J Med Case Reports 18 , 206 (2024). https://doi.org/10.1186/s13256-024-04519-x

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Received : 24 September 2023

Accepted : 24 March 2024

Published : 25 April 2024

DOI : https://doi.org/10.1186/s13256-024-04519-x

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