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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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• 04 December 2020
• Correction 09 December 2020

How to write a superb literature review

Andy Tay is a freelance writer based in Singapore.

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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

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

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• UConn Library
• Literature Review: The What, Why and How-to Guide
• Introduction

Literature Review: The What, Why and How-to Guide — Introduction

• Getting Started
• How to Pick a Topic
• Strategies to Find Sources
• Evaluating Sources & Lit. Reviews
• Tips for Writing Literature Reviews
• Writing Literature Review: Useful Sites
• Citation Resources
• Other Academic Writings

What are Literature Reviews?

So, what is a literature review? "A literature review is an account of what has been published on a topic by accredited scholars and researchers. In writing the literature review, your purpose is to convey to your reader what knowledge and ideas have been established on a topic, and what their strengths and weaknesses are. As a piece of writing, the literature review must be defined by a guiding concept (e.g., your research objective, the problem or issue you are discussing, or your argumentative thesis). It is not just a descriptive list of the material available, or a set of summaries." Taylor, D.  The literature review: A few tips on conducting it . University of Toronto Health Sciences Writing Centre.

Goals of Literature Reviews

What are the goals of creating a Literature Review?  A literature could be written to accomplish different aims:

• To develop a theory or evaluate an existing theory
• To summarize the historical or existing state of a research topic
• Identify a problem in a field of research 

Baumeister, R. F., & Leary, M. R. (1997). Writing narrative literature reviews .  Review of General Psychology , 1 (3), 311-320.

What kinds of sources require a Literature Review?

• A research paper assigned in a course
• A thesis or dissertation
• A grant proposal
• An article intended for publication in a journal

All these instances require you to collect what has been written about your research topic so that you can demonstrate how your own research sheds new light on the topic.

Types of Literature Reviews

What kinds of literature reviews are written?

Narrative review: The purpose of this type of review is to describe the current state of the research on a specific topic/research and to offer a critical analysis of the literature reviewed. Studies are grouped by research/theoretical categories, and themes and trends, strengths and weakness, and gaps are identified. The review ends with a conclusion section which summarizes the findings regarding the state of the research of the specific study, the gaps identify and if applicable, explains how the author's research will address gaps identify in the review and expand the knowledge on the topic reviewed.

• Example : Predictors and Outcomes of U.S. Quality Maternity Leave: A Review and Conceptual Framework:  10.1177/08948453211037398  

Systematic review : "The authors of a systematic review use a specific procedure to search the research literature, select the studies to include in their review, and critically evaluate the studies they find." (p. 139). Nelson, L. K. (2013). Research in Communication Sciences and Disorders . Plural Publishing.

• Example : The effect of leave policies on increasing fertility: a systematic review:  10.1057/s41599-022-01270-w

Meta-analysis : "Meta-analysis is a method of reviewing research findings in a quantitative fashion by transforming the data from individual studies into what is called an effect size and then pooling and analyzing this information. The basic goal in meta-analysis is to explain why different outcomes have occurred in different studies." (p. 197). Roberts, M. C., & Ilardi, S. S. (2003). Handbook of Research Methods in Clinical Psychology . Blackwell Publishing.

• Example : Employment Instability and Fertility in Europe: A Meta-Analysis:  10.1215/00703370-9164737

Meta-synthesis : "Qualitative meta-synthesis is a type of qualitative study that uses as data the findings from other qualitative studies linked by the same or related topic." (p.312). Zimmer, L. (2006). Qualitative meta-synthesis: A question of dialoguing with texts .  Journal of Advanced Nursing , 53 (3), 311-318.

• Example : Women’s perspectives on career successes and barriers: A qualitative meta-synthesis:  10.1177/05390184221113735

Literature Reviews in the Health Sciences

• UConn Health subject guide on systematic reviews Explanation of the different review types used in health sciences literature as well as tools to help you find the right review type
• << Previous: Getting Started
• Next: How to Pick a Topic >>
• Last Updated: Sep 21, 2022 2:16 PM
• URL: https://guides.lib.uconn.edu/literaturereview

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

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.

Prevent plagiarism, run a free check.

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.

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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Methodological Approaches to Literature Review

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The literature review can serve various functions in the contexts of education and research. It aids in identifying knowledge gaps, informing research methodology, and developing a theoretical framework during the planning stages of a research study or project, as well as reporting of review findings in the context of the existing literature. This chapter discusses the methodological approaches to conducting a literature review and offers an overview of different types of reviews. There are various types of reviews, including narrative reviews, scoping reviews, and systematic reviews with reporting strategies such as meta-analysis and meta-synthesis. Review authors should consider the scope of the literature review when selecting a type and method. Being focused is essential for a successful review; however, this must be balanced against the relevance of the review to a broad audience.

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Systematic Reviews in Educational Research: Methodology, Perspectives and Application

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Thomas, D., Zairina, E., George, J. (2023). Methodological Approaches to Literature Review. In: Encyclopedia of Evidence in Pharmaceutical Public Health and Health Services Research in Pharmacy. Springer, Cham. https://doi.org/10.1007/978-3-030-50247-8_57-1

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Literature Reviews

• What is a literature review?
• Steps in the Literature Review Process
• Define your research question
• Determine inclusion and exclusion criteria
• Choose databases and search
• Review Results
• Synthesize Results
• Analyze Results
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What is a Literature Review?

A literature or narrative review is a comprehensive review and analysis of the published literature on a specific topic or research question. The literature that is reviewed contains: books, articles, academic articles, conference proceedings, association papers, and dissertations. It contains the most pertinent studies and points to important past and current research and practices. It provides background and context, and shows how your research will contribute to the field. 

A literature review should: 

• Provide a comprehensive and updated review of the literature;
• Explain why this review has taken place;
• Articulate a position or hypothesis;
• Acknowledge and account for conflicting and corroborating points of view

From  S age Research Methods

Purpose of a Literature Review

A literature review can be written as an introduction to a study to:

• Demonstrate how a study fills a gap in research
• Compare a study with other research that's been done

Or it can be a separate work (a research article on its own) which:

• Organizes or describes a topic
• Describes variables within a particular issue/problem

Limitations of a Literature Review

Some of the limitations of a literature review are:

• It's a snapshot in time. Unlike other reviews, this one has beginning, a middle and an end. There may be future developments that could make your work less relevant.
• It may be too focused. Some niche studies may miss the bigger picture.
• It can be difficult to be comprehensive. There is no way to make sure all the literature on a topic was considered.
• It is easy to be biased if you stick to top tier journals. There may be other places where people are publishing exemplary research. Look to open access publications and conferences to reflect a more inclusive collection. Also, make sure to include opposing views (and not just supporting evidence).

Source: Grant, Maria J., and Andrew Booth. “A Typology of Reviews: An Analysis of 14 Review Types and Associated Methodologies.” Health Information & Libraries Journal, vol. 26, no. 2, June 2009, pp. 91–108. Wiley Online Library, doi:10.1111/j.1471-1842.2009.00848.x.

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Periodically, UT Libraries runs a workshop covering the basics and library support for literature reviews. While we try to offer these once per academic year, we find providing the recording to be helpful to community members who have missed the session. Following is the most recent recording of the workshop, Conducting a Literature Review. To view the recording, a UT login is required.

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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: 
• How to write a literature review faster with Paperpal? 
• 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  

1. 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. 

2. 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. 

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3. 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. 

4. 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. 

5. 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. 

6. 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. 

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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. 

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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. 

Whether you’re exploring a new research field or finding new angles to develop an existing topic, sifting through hundreds of papers can take more time than you have to spare. But what if you could find science-backed insights with verified citations in seconds? That’s the power of Paperpal’s new Research feature!  

How to write a literature review faster with Paperpal?

Paperpal, an AI writing assistant, integrates powerful academic search capabilities within its writing platform. With the Research feature, you get 100% factual insights, with citations backed by 250M+ verified research articles, directly within your writing interface with the option to save relevant references in your Citation Library. By eliminating the need to switch tabs to find answers to all your research questions, Paperpal saves time and helps you stay focused on your writing.   

Here’s how to use the Research feature:  

• Ask a question: Get started with a new document on paperpal.com. Click on the “Research” feature and type your question in plain English. Paperpal will scour over 250 million research articles, including conference papers and preprints, to provide you with accurate insights and citations. 
• Review and Save: Paperpal summarizes the information, while citing sources and listing relevant reads. You can quickly scan the results to identify relevant references and save these directly to your built-in citations library for later access. 
• Cite with Confidence: Paperpal makes it easy to incorporate relevant citations and references into your writing, ensuring your arguments are well-supported by credible sources. This translates to a polished, well-researched literature review. 

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 good literature review is an ongoing process, and it may be necessary to revisit and update it as your research progresses. By combining effortless research with an easy citation process, Paperpal Research streamlines the literature review process and empowers you to write faster and with more confidence. Try Paperpal Research now and see for yourself.  

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

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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. 

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The prevalence of psychosis has been shown to be disproportionately high amongst sexual and gender minority individuals. However, there is currently little consideration of the unique needs of this population in mental health treatment, with LGBTQA+ individuals facing barriers in accessing timely and non-stigmatising support for psychotic experiences. This issue deserves attention as delays to help-seeking and poor engagement with treatment predict worsened clinical and functional outcomes for people with psychosis. The present protocol describes the methodology for a scoping review which will aim to identify barriers and facilitators faced by LGBTQA+ individuals across the psychosis spectrum in help-seeking and accessing mental health support.

A comprehensive search strategy will be used to search Medline, PsycINFO, Embase, Scopus, LGBTQ+ Source, and grey literature. Original studies of any design, setting, and publication date will be included if they discuss barriers and facilitators to mental health treatment access and engagement for LGBTQA+ people with experiences of psychosis. Two reviewers will independently screen titles/abstracts and full-text articles for inclusion in the review. Both reviewers will then extract the relevant data according to pre-determined criteria, and study quality will be assessed using the Joanna Briggs Institute (JBI) critical appraisal checklists. Key data from included studies will be synthesised in narrative form according to the Guidance on the Conduct of Narrative Synthesis in Systematic Reviews.

The results of this review will provide a comprehensive account of the current and historical barriers and facilitators to mental healthcare faced by LGBTQA+ people with psychotic symptoms and experiences. It is anticipated that the findings from this review will be relevant to clinical and community services and inform future research. Findings will be disseminated through publication in a peer-reviewed journal and presented at conferences.

Scoping review registration

This protocol is registered in Open Science Framework Registries ( https://doi.org/10.17605/OSF.IO/AT6FC ).

Peer Review reports

The prevalence of psychotic disorders in the general population has been estimated to be around 0.27–0.75% [ 1 , 2 ], with the lifetime prevalence of ever having a psychotic experience being estimated at 5.8% [ 3 ]. However, rates of psychotic symptoms and experiences are disproportionately high amongst LGBTQA+ populations, with non-heterosexual individuals estimated to be 1.99–3.75 times more likely to experience psychosis than their heterosexual peers [ 4 , 5 , 6 , 7 ]. Additionally, it has been estimated that transgender or gender non-conforming (henceforth trans) individuals are 2.46–49.7 times more likely than their cisgender peers (i.e. individuals whose gender identity is the same as their birth registered sex) to receive a psychotic disorder diagnosis [ 8 , 9 ]. The increased rates of psychotic experiences noted amongst gender and sexual minorities may be explained by evidence indicating that LGBTQA+ people are also exposed to risk factors for psychosis at a far greater rate than members of the general population, such as childhood adversity [ 10 , 11 , 12 ], minority stress [ 13 ], discrimination [ 14 ], and stigma [ 15 , 16 ]. Furthermore, there is added potential for diagnostic biases leading to over-diagnosing psychosis in gender diverse individuals, whose gender expression and dysphoria may be pathologized by mental health service providers [ 8 ].

Despite these concerning statistics, there is very little research examining the experiences of LGBTQA+ people with psychosis, and limited consideration of the unique needs these individuals may have in accessing and engaging with mental health services. While timely access to treatment has consistently been associated with better symptomatic and functional outcomes for people with psychosis [ 17 , 18 ], there are often delays to treatment initiation which are worsened for LGBTQA+ individuals [ 19 , 20 ]. These individuals face additional barriers to accessing adequate mental health support compared to cisgender/heterosexual people [ 19 ] and may need to experiment with several mental health services before finding culturally competent care [ 20 ]. This in turn may lead to longer duration of untreated psychosis. Additionally, there seems to be a lack of targeted support for this population from healthcare providers, with LGBTQA+ individuals with serious mental health concerns reporting higher rates of dissatisfaction with psychiatric services than their cisgender and heterosexual counterparts [ 7 , 14 , 21 ]. However, the extent of these differences varies across contexts [ 22 ], potentially due to improved education around stigma and LGBTQA+ issues within a subset of mental health services.

Nonetheless, stigma remains one of the highest cited barriers to help-seeking for mental health problems, particularly with regard to concerns around disclosure [ 23 ], which can be particularly challenging for people experiencing psychosis [ 24 , 25 ]. Stigma stress in young people at risk for psychosis is associated with less positive attitudes towards help-seeking regarding both psychiatric medication and psychotherapy [ 26 ], potentially partly due to fears of judgement and being treated differently by service providers [ 27 ]. This issue may be compounded for people who also belong to minoritized groups [ 23 , 28 ], particularly as LGBTQA+ individuals have reported experiencing frequent stigma and encountering uninformed staff when accessing mental healthcare [ 7 , 29 ]. Furthermore, stigma-fuelled hesitance to access services may be heightened for trans people [ 30 ] whose identities have historically been pathologized and conflated with experiences of psychosis [ 31 ].

Even when individuals manage to overcome barriers to access support, there are added challenges to maintaining adequate treatment engagement. In a large online study, half of trans and nearly one third of LGB participants reported having stopped using mental health services in the past because of negative experiences related to their gender identity or sexuality [ 20 ]. This can be particularly problematic as experiences of stigma predict poorer medication adherence in psychosis [ 32 ] which subsequently multiplies the risk for relapse and suicide [ 33 ]. While no research to date has explored non-adherence rates in people with psychosis who are LGBTQA+, concerns around suicidality are heightened for individuals who are gender and sexuality diverse [ 34 , 35 , 36 ].

Generally, there is rising demand for mental healthcare that specifically addresses the needs of gender and sexual minority individuals and promotes respect for diversity, equity, and inclusion [ 29 , 37 ]. This is particularly salient as positive relationships with staff are associated with better medication adherence for people with psychosis [ 38 ] and healthcare providers with LGBTQA+-specific mandates have demonstrated higher satisfaction rates for LGBTQA+ individuals [ 20 ]. Mental health services need to adapt treatment options to acknowledge minority stress factors for those with stigmatised identities and, perhaps more importantly, how these intersect and interact to increase inequalities in people from minoritized groups accessing and benefiting from treatment [ 37 , 39 ].

Additionally, gender affirming care needs to be recognised as an important facet of mental health treatment for many trans individuals, as it is associated with positive outcomes such as improvements in quality of life and psychological functioning [ 40 , 41 , 42 ] and reductions in psychiatric symptom severity and need for subsequent mental health treatment [ 8 , 43 ]. While there are additional barriers in access to gender affirming care for individuals with psychosis, this treatment has shown success in parallel with treatment to address psychosis symptom stabilisation [ 19 , 44 ]. The importance of affirmation is echoed by the finding that many negative experiences of LGBTQA+ participants with mental health services could be avoided simply by respecting people’s pronouns and using gender-neutral language [ 20 ].

To ensure timely access to appropriate treatment for LGBTQA+ people with psychosis, there is a need for improved understanding of the factors which challenge and facilitate help-seeking and engagement with mental health support. A preliminary search of Google Scholar, Medline, the Cochrane Database of Systematic Reviews, and PROSPERO was conducted and revealed no existing or planned reviews exploring benefits and/or obstacles to mental health treatment specific to this population. Therefore, the proposed review seeks to comprehensively search and appraise the existing literature to identify and summarise a range of barriers and facilitators to adequate mental health support faced by LGBTQA+ people with experiences of psychosis. This will allow for the mapping of the types of evidence available and identification of any knowledge gaps. Moreover, we hope to guide future decision-making in mental healthcare to improve service accessibility for LGBTQA+ individuals with psychosis and to set the foundations for future research that centres this marginalised population. Based on published guidance [ 45 , 46 , 47 ], a scoping review methodology was identified as the most appropriate approach to address these aims.

Selection criteria

This scoping review protocol has been developed in compliance with the JBI Manual for Evidence Synthesis [ 48 ] and, where relevant, the Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) checklist [ 49 ] (see Additional file 1). In the event of protocol amendments, the date, justification, and description for each amendment will be provided.

Due to the limited literature around the topic of this review, any primary original study design, setting, and publication date will be considered for inclusion. Publications written in English will be included, and articles in other languages may be considered pending time and cost constraints around translation. Publications will be excluded if the full text is not available upon request from authors.

The PCC (Population, Concept, Context) framework was used to develop the inclusion criteria for this scoping review:

This review will include individuals of any age who are LGBTQA+ and have had experiences of psychosis. For the purposes of this review, ‘LGBTQA+ individuals’ will be broadly defined as any individual that is not heterosexual and/or cisgender or anyone who engages in same-gender sexual behaviour. Studies may include participants who are cisgender and heterosexual if they separately report outcomes for LGBTQA+ individuals. Within this review, the term ‘psychosis’ includes (i) any diagnosis of a psychotic disorder, such as schizophrenia spectrum disorders, mood disorders with psychotic features, delusional disorders, and drug-induced psychotic disorders, (ii) sub-threshold psychotic symptoms, such as those present in ultra-high risk (UHR), clinical high risk (CHR), or at risk mental state (ARMS) individuals, and (iii) any psychotic-like symptoms or experiences. Studies may include participants with multiple diagnoses if they separately report outcomes for individuals on the psychosis spectrum.

This review will include publications which discuss potential barriers and/or facilitators to mental health help-seeking and/or engagement with mental health treatment. ‘Barriers’ will be operationalised as any factors which may delay or prevent individuals from accessing and engaging with appropriate mental health support. These may include lack of mental health education, experienced or internalised stigma, experiences of discrimination from health services, and lack of inclusivity in health services. ‘Facilitators’ will be operationalised as any factors which may promote timely help-seeking and engagement with sources of support. These may include improved access to mental health education, positive sources of social support, and welcoming and inclusive services. Mental health help-seeking will be broadly defined as any attempt to seek and access formal or informal support to address a mental health concern related to experiences of psychosis (e.g. making an initial appointment with a service provider, seeking help from a friend). Mental health treatment engagement will be broadly defined as adherence and active participation in the treatment that is offered by a source of support (e.g. attending scheduled appointments, taking medication as prescribed, openly communicating with service providers).

This review may include research encompassing any setting in which mental healthcare is provided. This is likely to include formal healthcare settings such as community mental health teams or inpatient clinics as well as informal settings such as LGBTQA+ spaces or informal peer support. Studies will be excluded if they focus exclusively on physical health treatment.

Search strategy

Database searches will be conducted in Medline, PsycINFO, Embase, Scopus, and LGBTQ+ Source. The full search strategy for this protocol is available (see Additional file 2). This strategy has been collaboratively developed and evaluated by a scholarly services health librarian. Searches will include subject headings relevant to each database and title/abstract keywords relating to three main concepts: (i) LGBTQA+ identity, (ii) experiences of psychosis, and (iii) mental health treatment. Keywords for each concept will be combined using the Boolean operator ‘OR’, and the three concepts will be combined using ‘AND’. This search strategy was appropriately translated for each of the selected databases. There will be no limitations on language or publication date at this stage to maximise the breadth of the literature captured. Publications returned from these searches will be exported to EndNote. Searches will be re-run prior to the final analysis to capture any newly published studies.

The database searches will be supplemented by searching the grey literature as per the eligibility criteria detailed above. These may include theses and dissertations, conference proceedings, reports from mental health services, and policy documents from LGBTQA+ groups. Google and Google Scholar will be searched using a combination of clauses for psychosis (Psychosis OR psychotic OR schizophrenia OR schizoaffective), treatment (treatment or “help-seeking”), and queer identity. The latter concept will have three clauses for three separate searches, with one including broad queer identity (LGBT), one specific to non-heterosexual individuals (gay OR lesbian OR homosexual OR bisexual OR queer OR asexual), and one specific to trans individuals (transgender OR transsexual OR transexual OR “non-binary” OR “gender minority”). Additionally, reference lists and citing literature will be manually searched for each paper included in the review to capture any articles and policy documents not previously identified.

Data selection

Search results will be imported into Covidence using EndNote, and duplicates will be eliminated. Titles and abstracts will be screened by the first and second authors according to pre-defined screening criteria, which will be discussed by the authors and piloted prior to screening. These criteria will consider whether the articles included LGBTQA+ participants with experiences of psychosis (as operationalised above) in relation to mental health help-seeking and/or treatment. Full texts of relevant articles will then be obtained and screened by the first and second reviewer in accordance with the full inclusion and exclusion criteria after initial piloting to maximise inter-rater reliability. Decisions on inclusion and exclusion will be blinded and recorded on Covidence. Potential discrepancies will be resolved through discussion, and when consensus cannot be reached, these will be resolved by the supervising author. The process of study selection will be documented using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) flow diagram [ 50 ].

Data extraction

Data extraction will be performed independently by two reviewers using Covidence. Prior to beginning final extraction, both reviewers will independently pilot the extraction tool using a sample of five included studies and discuss any necessary changes. Information extracted is planned to include the following: title, author name(s), year of publication, country in which the study was conducted, study design, sample size, population of focus (i.e. sexual minorities, gender minorities, or both), sample demographics (i.e. age, gender identity, and sexual orientation), setting (e.g. early intervention service, community mental health team, etc.), psychosis characteristics (e.g. diagnoses included, severity of symptoms, etc.), type of treatment (e.g. cognitive behavioural therapy, antipsychotic medication, etc.), and any barriers and/or facilitators identified according to the aforementioned operationalised definitions. Disagreements will be resolved through discussion between the two reviewers and, when necessary, final decisions will be made by a senior supervisor. Once extracted, information will be recorded in Excel. Lead authors of papers will be contacted by the primary review author in cases where there is missing or insufficient data.

Quality assessment

Due to the expected heterogeneity in the types of studies that may be included in this review (e.g. qualitative studies, randomised controlled trials, case control studies, case reports), the relevant revised Joanna Briggs Institute (JBI) critical appraisal checklists [ 51 ] will be used to assess risk of bias and study quality for each study design. Two reviewers will independently use these checklists to assess each paper that is included following the full-text screening. If there are discrepancies in article ratings, these will be resolved through discussion between the two authors. If no consensus is reached, discrepancies will be resolved by a senior supervisor. In line with the scoping nature of this review, low-quality studies will not be excluded from the synthesis.

Evidence synthesis

Data from included studies will be synthesised using a narrative synthesis approach in accordance with the Guidance on the Conduct of Narrative Synthesis in Systematic Reviews [ 52 ]. A preliminary descriptive synthesis will be conducted by tabulating the extracted data elements from each study alongside quality assessment results and developing an initial description of the barriers and facilitators to (1) accessing and (2) engaging with mental health support that are identified in the literature. This initial synthesis will then be interrogated and refined to contextualise these barriers and facilitators in the setting, population, and methodology of each study to form the basis for an interpretative synthesis.

This review will not use a pre-existing thematic framework to categorise barriers and facilitators as it is expected that the factors identified will not neatly fit into existing criteria. Instead, these will be conceptualised according to overarching themes as interrelated factors, so that potentially complex interactions between barriers and facilitators within and across relevant studies may be explored through concept mapping. If most of the studies included are qualitative, there may also be scope for a partial meta-synthesis. To avoid oversimplifying the concept of ‘barriers and facilitators’ (see criticism by Bach-Mortensen & Verboom [ 53 ]), this data synthesis will be followed by a critical reflection of the findings through the lens of the socio-political contexts which may give rise to the barriers and facilitators identified, exploring the complexities necessary for any changes to be implemented in mental health services.

If the extracted data indicate that gender minority and sexual minority individuals experience unique or different barriers and/or facilitators to each other, these population groups will be analysed separately as opposed to findings being generalised across the LGBTQA+ spectrum. Furthermore, if there is scope to do so, analyses may be conducted to investigate how perceived barriers and facilitators for this population may have changed over time (i.e. according to publication date) as definitions of psychosis evolve and LGBTQA+ individuals gain visibility in clinical services.

The proposed review will add to the literature around mental health treatment for LGBTQA+ people with psychosis. It will provide a thorough account of the barriers and facilitators to accessing and engaging with support faced by this population and may inform future research and clinical practice.

In terms of limitations, this review will be constrained by the existing literature and may therefore not be sufficiently comprehensive in reflecting the barriers and facilitators experienced by subgroups within the broader LGBTQA+ community. Additionally, although broad inclusion criteria are necessary to capture the full breadth of research conducted in this topic, included studies are likely to be heterogeneous and varied in terms of their methodology and population which may complicate data synthesis.

Nonetheless, it is anticipated that the findings from this review will provide the most comprehensive synthesis to date of the issues driving low help-seeking and treatment engagement in people across the psychosis spectrum who are LGBTQA+. This review will likely also identify gaps in the literature which may inform avenues for future research, and the factors identified in this review will be considered in subsequent research by the authors.

Additionally, findings will be relevant to healthcare providers that offer support to people with psychosis who may have intersecting LGBTQA+ identities as well as LGBTQA+ organisations which offer support to LGBTQA+ people who may be experiencing distressing psychotic experiences. These services are likely to benefit from an increased awareness of the factors which may improve or hinder accessibility for these subsets of their target populations. Therefore, results from this review may inform decision-making around the implementation of service-wide policy changes.

The findings of this review will be disseminated through the publication of an article in a peer-reviewed journal and presented at relevant conferences in Australia and/or internationally. Additionally, the completed review will form part of the lead author’s doctoral thesis.

Availability of data and materials

Not applicable for this protocol.

Abbreviations

• At risk mental state

Clinical high risk for psychosis

Joanna Briggs Institute

Lesbian, gay, and bisexual

Lesbian, gay, bisexual, transgender, queer or questioning, asexual or aromantic, and more

Population, Concept, Context

Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols

Ultra-high risk for psychosis

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Acknowledgements

The authors would like to acknowledge the support of Ms Olivia Larobina, Scholarly Services Librarian (STEMM) at Deakin University, in the development of the search strategy.

CCG is funded by a Deakin University Postgraduate Research (DUPR) Scholarship. ZW is funded by a University of Western Australia Research Training Program (RTP) Scholarship. AL is supported by a National Health and Medical Research Council (NHMRC) Emerging Leaders Fellowship (2010063). LJW is supported by a NHMRC Emerging Leaders Fellowship (1174060). ARY is supported by a NHMRC Principal Research Fellowship (1136829). The funding providers had no role in the design and conduct of the study, or in the preparation, review, or approval of this manuscript.

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Additional file 1. prisma-p 2015 checklist. completed prisma-p checklist for this systematic review protocol., 13643_2024_2566_moesm2_esm.docx.

Additional file 2. Search Strategy. Detailed search strategy for this systematic review, including search terms and relevant controlled vocabulary terms for each included database.

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Surgical intervention of Lemierre’s syndrome: a case report and review of the literature

• Yiqi Pan 1   na1 ,
• Zhihong Shi 1   na1 ,
• Qian Da 2 ,
• Chaofu Wang 2 ,
• Yilin Shen 1 &
• Mingliang Xiang   ORCID: orcid.org/0000-0001-9253-9852 1  

Journal of Medical Case Reports volume  18 , Article number:  265 ( 2024 ) Cite this article

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Lemierre’s syndrome is a fatal and rare disease that is typically characterized by oropharyngeal infection and internal jugular vein thrombosis. Timely institution of appropriate antibiotics is the standard treatment.

Case presentation

The authors report a case of Lemierre’s syndrome. A 67-year-old male patient of Han ethnicity in China suffered from a large inflammatory neck mass involving left internal jugular vein thrombosis diagnosed as Lemierre’s syndrome and finally cured by surgical treatment. In addition, a literature review was carried out through PubMed using the terms “Lemierre’s syndrome/disease and review, meta-analysis or retrospective study” and “Lemierre’s syndrome/disease and internal jugular vein”. This search yielded six articles that recorded surgical methods such as drainage, craniotomy, tooth extraction, and ligation of the occluded vein to give clinicians more ideas about the treatment of the Lemierre’s syndrome.

This is the first review to summarize the conditions under which surgical treatment are conducted. Additionally, this is the first report of such a large inflammatory neck mass that was completely cured by surgical resection and internal jugular vein ligation. The authors also offer several conclusions regarding surgical intervention in Lemierre’s syndrome for the first time.

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Internal jugular vein (IJV) thrombosis is a relatively rare and urgent disease. In a retrospective study, the number of such cases occurring from 2001 to 2008 was 2.5 times that occurring from 1991 to 2000 and 20 times that occurring from 1980 to 1990 [ 1 ]. The reasons behind the increasing incidence of IJV thrombosis include the increase in antibiotic resistance, widespread use of hemodialysis, general application of central venous catheters, expansion of assisted reproductive technology, and increasing incidence of cancer [ 2 ]. In a 9-year retrospective study of 1948 patients with deep vein thrombosis, only 29 patients developed IJV thrombosis, of whom 23 had IJV thrombosis secondary to another condition, such as a malignancy (for example, Trousseau syndrome), central venous catheter implantation, or ovarian hyperstimulation syndrome (OHSS) [ 3 ]. In addition, bilateral internal jugular vein thrombosis is an important indicator of malignant tumors. In a 5-year retrospective study of 41 patients with IJV thrombosis in Germany, paraneoplastic thrombosis accounted for 54% of cases; of these cases, otolaryngology head and neck diseases accounted for 68%. The other patients mostly had inflammatory diseases [ 4 ].

In the ear, nose, and throat (ENT) field, IJV thrombosis is commonly associated with Lemierre’s syndrome (LS), which is a complication of infectious diseases, such as otitis media and oropharyngeal abscess or infection. LS is commonly defined by the following diagnostic criteria: (1) oropharyngeal infection; (2) internal jugular vein thrombophlebitis or thrombosis; (3) septic emboli at a remote site, more frequently the lungs; and (4) isolation of Fusobacterium nucleatum on blood culture [ 5 ]. LS is usually accompanied by septic emboli in the lungs or other organs [ 6 ]. Under some rare conditions, LS can also be triggered by tooth extraction [ 7 ]. Pulmonary embolism, with an incidence of approximately 10%, and postthrombotic syndromes, such as limb pain, heaviness, venous dilatation, edema, pigmentation, nutritional skin changes, and venous ulcers, are complications of IJV thrombosis [ 4 ]. Therefore, ENT doctors should give enough attention to patients with IJV thrombosis to avoid the disastrous results caused by pulmonary or cerebral thrombosis.

To the best of our knowledge, this is the first case of such a large infectious neck mass with internal jugular vein thrombosis that was completely cured by surgical intervention.

A 67-year-old Chinese male of Han ethnicity developed pain in the left neck 14 days prior after eating mud fish. He was healthy and denied a history of infectious diseases, chronic diseases, thrombotic disease, surgical trauma, blood transfusion, allergies, or contact with poisonous substances. Physical examination on admission revealed the following: fever, chills, fatigue, mild dysuria, diffusive swelling pain of the neck on the left side, and high skin temperature. His left neck was tender and edematous with cellulitis. The mass was scleroid with a liquefied center. Other parameters were as follows: white blood cell (WBC) count, 12.2 × 10 9 /L; neutrophil count, 11.76 × 10 9 /L; platelet (PLT) count, 51 × 10 9 /L; C-reactive protein (CRP) level, 146 mg/L; procalcitonin (PCT), 156.99 ng/mL; temperature, 39.5 °C; respiratory rate, 24 breaths per minute; pulse, 118 beats per minute; and blood pressure (BP), 109/61 mmHg. The patient stated that he had been to many hospitals in the last 2 weeks and that the use of antibiotics such as ceftriaxone and metronidazole slightly alleviated his neck pain at first. However, the effect was temporary and no longer present after he transferred to Shanghai, and 3 days before he presented to our hospital, he noticed extreme swelling and felt increased pain in the area of his neck mass (Fig.  1 ). A series of further examinations were performed, with the following results: glucose, 22.13 mmol/L; activated partial thromboplastin time (APTT), 43.1 seconds; prothrombin time (PT), 17.3 seconds; fibrinogen (Fg), 5.0 g/L; fibrin/fibrinogen degradation products (FDP), 7.3 mg/L; and D-dimer (D-D), 2.1 mg/L. Urine analysis was positive for glucose, blood, protein, and white blood cells. Infectious diseases and acute nephrology were considered. The culture of blood and fluid obtained from the mass was negative for any bacteria including anaerobic bacteria, mycoplasma, or fungus. Ultrasound revealed mixed echogenicity in the left neck mass that was irregular in shape. The mass was approximately 74 × 37 mm in size. Color Doppler revealed generalized thrombosis of the internal jugular vein. CT of the chest and neck was conducted and suggested that the cervical abscess extended to the thorax and superior mediastinum, without a signal from the left internal jugular vein. Multiple enlarged cervical lymph nodes were observed. The trachea and left thyroid were also compressed (Fig.  2 ). Video laryngoscopy excluded the possibility of pyriform sinus fistula or any foreign body. Pus obtained from the mass showed many neutrophils and large amounts of necrotic tissue, and 14 days of combination antibiotic treatment (imipenem and teicoplanin) and regular insulin therapy in our hospital returned the patient’s temperature, routine blood markers, CRP and PCT levels, and coagulation function to normal. However, the neck mass remained. Therefore, the surgery department was consulted.

Picture of the patient’s neck showed a huge mass with tenderness (red circles)

A Coronal plane of computed tomography showed neck mass spread down to superior mediastinum with iquefactive necrosis in the center (red arrows). Trachea was oppressed to the right side. B Significant intraluminal filling defect and thrombosis was found in internal jugular vein (yellow arrow)

The patient was taken to the operating room. The neck mass adhered tightly to the surrounding tissue, and the involved segment of the left internal jugular vein was exposed by sharp dissection (Fig.  3 A). The proximal part of the mass needed to be ligated first to avoid small thrombus detachment. There was a large amount of inflammation and fibrosis present in the involved area. The involved segment of the internal jugular vein and the whole neck mass were completely resected, and the distal part of the internal jugular vein was fully ligated (Fig.  3 B). The pathological examination showed hemorrhagic necrosis with the proliferation of fibrous and granulation tissue and the accumulation of foam-like cells and multinuclear giant cells (Fig.  4 ). The patient recovered and was discharged a week after surgery. No other adverse events happened.

Intraoperative view showed ligation of internal jugular vein and separation of the mass ( A ). The resected neck mass as well as left internal jugular vein was shown. Intraluminal thrombosis could be seen clearly when opening the internal jugular vein ( B )

Representative pathological photomicrograph demonstrated thrombus ( A ) and abscess formation ( B )

Literature review

It is rare that patients with Lemierre’s syndrome require surgical intervention when antibiotics and anticoagulant therapies fail. This is the first study to summarize cases of LS requiring surgical treatment (not including abscess drainage). First, we decided to collect previous reviews and meta-analyses to obtain good knowledge of the rate of surgery in LS. Search strategy and selection criteria were as follows. A search of the literature in MEDLINE was performed through PubMed to identify relevant English language articles from 1980 to 2022. The following search terms were used: “Lemierre’s syndrome/disease and review, meta-analysis or retrospective study” and “Lemierre’s syndrome/disease and internal jugular vein”. The references of the retrieved articles were also reviewed to identify additional sources. Through reading the abstract and the full text, we found a total of six reviews and meta-analyses (Table  1 ) that included detailed descriptions of patients who underwent surgery (not including abscess drainage). In a retrospective review from 1998 to 2010 at a local tertiary referral hospital, 17 of the 23 patients underwent surgical treatment of the primary infection site [ 8 ]. In a 5-year systematic review, surgical procedures, such as tooth extraction, craniotomy, and ligation of the occluded vein, were performed in five patients to prevent further septic emboli [ 9 ]. A retrospective study from June 2000 to May 2016 showed that IJV ligation was performed in only one of five LS cases at the Children’s Hospital of Alabama [ 10 ]. In an 8-year Swedish nationwide retrospective study, three patients with peritonsillitis were surgically treated by tonsillectomy [ 11 ]. In the latest meta-analysis of 394 patients in 2020, only 10 patients underwent IJV ligation/excision, only 1 patient underwent ligation/excision of the thrombosed external carotid artery, 3 underwent endoscopic sinus surgery, and 11 underwent mastoidectomy [ 12 ]. In the latest systematic review, which included the most LS cases reported, surgical procedures were performed in 101 patients, and 31 patients underwent IJV ligation/embolectomy [ 13 ].

In this study, we report a case of Lemierre’s syndrome in an elderly male caused by an acute infectious neck mass. Timely comprehensive medical and surgical treatments were given to avoid serious complications.

Internal jugular vein thrombosis is a rare and serious emergent disease that needs to be identified early in the course, as it can lead to catastrophic consequences, such as stroke or pulmonary embolism. The main pathological basis of internal jugular vein thrombosis is as follows: (1) injury of venous intima; (2) slowing down of blood flow; and (3) hypercoagulability. The common causes are as follows [ 14 , 15 , 16 ]: (1) facial infection, such as furuncle and carbuncle, sinusitis, otitis media, and suppurative tonsillitis; bacteria can spread through the damaged mucosa and parapharyngeal space or invade the jugular vein through the lymphatic and venous systems, leading to infectious phlebitis and bacterial embolism information; (2) long duration of internal jugular vein catheterization; (3) head and neck surgery; (4) head and neck tumor; (5) pulmonary embolism; and (6) other systemic diseases, such as polycythemia. Doctors need to take care of patients immediately when encountering such cases.

Lemierre’s syndrome can show the typical symptoms and signs of progressive infection, including sore throat, fever, or neck pain. A systematic review of Lemierre’s syndrome by Peter et al . found that in 84 patients, the most common first clinical presentation was a sore throat (33%), followed by a neck mass (23%) and neck pain (20%) [ 1 ]. In the current case, the patient presented with fever and neck pain at first, followed by a neck mass. The use of antibiotics before he was transferred to our hospital was ineffective. The white blood cell count, PCT level, and erythrocyte sedimentation rate (ESR) were elevated, and blood appeared in his urine. We adjusted the treatment to the combined application of broad-spectrum antibiotics, including imipenem and teicoplanin, for another 2 weeks. The patient’s body temperature returned to normal and laboratory testing showed that the patient’s infectious condition had been controlled, but the neck mass and internal jugular vein thrombosis persisted and required surgical treatment.

Fusobacterium necrophorum is the main pathogen of Lemierre’s syndrome [ 1 ]. However, in this case, the culture of both blood and fluid obtained from the mass was negative for bacteria, which might be because the patient had been treated with antibiotics (mainly including ceftriaxone and metronidazole) for nearly 2 weeks before coming to our hospital. This also suggests that it is particularly important for doctors to culture blood or fluid from the mass before any use of antibiotics in these patients. In many reviews, a large proportion of the cases also did not report any microbiological agent [ 9 , 12 , 13 ] and thus far a clinical diagnosis of LS is still valid if the bacteria go undetected. As for the reason about how this patient got such infectious disease, considering that he had eaten mud fish before the onset of disease and CT showed that mucous of left pyriform sinus was edematous and enhanced (Fig.  5 ), we considered that neck infection might be secondary to the infection of mucous scratch in left pyriform sinus, and uncontrolled hyperglycemia was an important factor to cause serious infection.

Coronal plane of computed tomography showed disappearance of left pyriform sinus surrounding by abnormal enhancement (yellow arrow)

The diagnosis of internal jugular vein thrombosis in Lemierre’s syndrome relies on imaging examination. Ultrasound is the first choice for the diagnosis of LS, and CT and magnetic resonance imaging (MRI) are currently implemented in general practice when necessary. Albertyn et al . first summarized the classic imaging features of internal jugular vein thrombosis. Ultrasonography shows the vein to be distended and nonpulsatile, with internal echoes. CT shows swelling of the adjacent soft tissues, distension of the vein with wall enhancement, and low-attenuation intraluminal filling defects. However, ultrasound has limitations and cannot display the anatomy behind the clavicle or mandible [ 16 ]. In this case, we found that although US can clearly show internal jugular vein thrombosis, knowing its boundaries and connection with the tumor still depends on CT or MRI, especially when there is an urgent need for surgery. A full assessment by preoperative imaging is of great importance. This is also consistent with the views of Charles et al . [ 17 ].

Priority treatment for LS includes antibiotic therapy and drainage of the infected site. Here in our hospital, considering that cephalosporins such as ceftriaxone did not control the fever and pain in this patient, imipenem and teicoplanin were used according to experience to cover wide varieties of Gram-negative and Gram-positive bacteria, including both anaerobic and aerobic bacteria. In addition, teicoplanin has lower side effects than vancomycin [ 18 ]. Rarely, other surgical procedures, such as ligation of the occluded vein, craniotomy, and tooth extraction, are performed. Antithrombotic therapy, including novel oral anticoagulants (DOACs), is also recommended depending on the individual’s condition. However, it remains controversial whether anticoagulation or antithrombotics are effective in Lemierre’s syndrome. Some scholars think that thrombosis is due to the infection process and can be resolved when the infection has been controlled [ 19 ]. In this case, we did not immediately apply anticoagulant or thrombolytic therapies considering that the APTT of this patient was significantly prolonged at the time he came to our hospital and the consumption of platelets was relatively high; emergency anticoagulant therapy may have increased his risk of bleeding. To date, there have been no sufficient clinical studies and no sufficient evidence suggesting the necessity for anticoagulant therapy in Lemierre’s syndrome [ 20 ]. Previous studies have also reported the occurrence of extensive suppurative thrombophlebitis of the bilateral IJV and superior vena cava in patients with Lemierre’s syndrome despite the use of antibiotics and anticoagulant therapy; adjunctive catheter-directed thrombolysis and superior vena cava stenting were performed to help these patients completely recover [ 21 ]. Anticoagulation therapy has not been shown to reduce the complications of Lemierre’s syndrome, such as sepsis [ 17 ]. Meanwhile, Johannesen et al . did not find that anticoagulation therapy decreased the mortality rate or course of the disease or reduced the duration of antibiotic use [ 9 ]. However, anticoagulation therapy is recommended in patients with a poor clinical response despite antibiotic therapy and with a high risk of intracranial thrombosis or recurrent thrombophlebitis [ 22 , 23 , 24 ]. In this case, the cause of internal jugular vein thrombosis was largely infection, so surgical treatment was the best choice when antibiotics could not completely cure the infection and thrombosis. Through previous retrospective studies, systematic reviews and meta-analyses obtained by database searches, we summarized the following points regarding surgical intervention in Lemierre’s syndrome:

When patients do not respond to conservative medical therapy and continue to show extensive septic thrombosis or uncontrolled severe sepsis, surgical treatments need to be considered.

Abscess drainage is the most common and convenient surgical treatment for abscesses upon formation.

Surgical treatment of the primary infection site is effective for controlling the spread of infection and sepsis.

IJV ligation or excision is suitable for patients with persistent septic embolization after treatment with antibiotics and anticoagulants.

IJV ligation or excision is also appropriate to avoid thrombus detachment when anticoagulation therapy or catheter-directed thrombolysis is ineffective.

Lemierre’s syndrome is an extremely rare disease, but the fatality rate can reach 15%, even with escalating antibiotic therapy [ 21 ] . Therefore, early diagnosis is particularly important, and the timely institution of appropriate antibiotics is the standard treatment. Surgical intervention may be the only effective option for controlling the source of infection or when conservative medical treatment fails.

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Abbreviations

Internal jugular vein

Ear, nose, and throat

• Lemierre’s syndrome

White blood cell

C-reactive protein

Procalcitonin

Blood pressure

Computed tomography

Magnetic resonance imaging

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Acknowledgements

Not applicable.

This study was supported by the National Natural Science Foundation of China (grant nos. 82101212, 82101209, 82301296, 82301297), Science and Technology Commission of Shanghai Municipality (grant nos. 23ZR1440200, 21ZR1440200, SHDC2020CR1044B-003), Shanghai “Rising Stars of Medical Talents” Youth Development Program, and Shanghai Municipal Hospital ENT Specialist Alliance.

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Yiqi Pan and Zhihong Shi contributed equally to this work.

Authors and Affiliations

Department of Otolaryngology and Head and Neck Surgery, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China

Yiqi Pan, Zhihong Shi, Bin Ye, Yilin Shen & Mingliang Xiang

Department of Pathology, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China

Qian Da & Chaofu Wang

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Contributions

Mingliang Xiang performed the whole operation and checked the final review. Yilin Shen revised and reviewed the original manuscript. Yiqi Pan wrote the whole manuscript. Zhihong Shi took part in the whole operation and carried out the literature review. Bin Ye reviewed the manuscript. Chaofu Wang and Qian Da provided the pathological images. All authors read and approved the final manuscript.

Corresponding authors

Correspondence to Yilin Shen or Mingliang Xiang .

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The studies involving human participants were reviewed and approved by Human Ethics Committee, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine. The patient provided his written informed consent for the use of his images and other clinical information in this study. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

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

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Pan, Y., Shi, Z., Ye, B. et al. Surgical intervention of Lemierre’s syndrome: a case report and review of the literature. J Med Case Reports 18 , 265 (2024). https://doi.org/10.1186/s13256-024-04584-2

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Lau F, Kuziemsky C, editors. Handbook of eHealth Evaluation: An Evidence-based Approach [Internet]. Victoria (BC): University of Victoria; 2017 Feb 27.

Handbook of eHealth Evaluation: An Evidence-based Approach [Internet].

Chapter 9 methods for literature reviews.

Guy Paré and Spyros Kitsiou .

9.1. Introduction

Literature reviews play a critical role in scholarship because science remains, first and foremost, a cumulative endeavour ( vom Brocke et al., 2009 ). As in any academic discipline, rigorous knowledge syntheses are becoming indispensable in keeping up with an exponentially growing eHealth literature, assisting practitioners, academics, and graduate students in finding, evaluating, and synthesizing the contents of many empirical and conceptual papers. Among other methods, literature reviews are essential for: (a) identifying what has been written on a subject or topic; (b) determining the extent to which a specific research area reveals any interpretable trends or patterns; (c) aggregating empirical findings related to a narrow research question to support evidence-based practice; (d) generating new frameworks and theories; and (e) identifying topics or questions requiring more investigation ( Paré, Trudel, Jaana, & Kitsiou, 2015 ).

Literature reviews can take two major forms. The most prevalent one is the “literature review” or “background” section within a journal paper or a chapter in a graduate thesis. This section synthesizes the extant literature and usually identifies the gaps in knowledge that the empirical study addresses ( Sylvester, Tate, & Johnstone, 2013 ). It may also provide a theoretical foundation for the proposed study, substantiate the presence of the research problem, justify the research as one that contributes something new to the cumulated knowledge, or validate the methods and approaches for the proposed study ( Hart, 1998 ; Levy & Ellis, 2006 ).

The second form of literature review, which is the focus of this chapter, constitutes an original and valuable work of research in and of itself ( Paré et al., 2015 ). Rather than providing a base for a researcher’s own work, it creates a solid starting point for all members of the community interested in a particular area or topic ( Mulrow, 1987 ). The so-called “review article” is a journal-length paper which has an overarching purpose to synthesize the literature in a field, without collecting or analyzing any primary data ( Green, Johnson, & Adams, 2006 ).

When appropriately conducted, review articles represent powerful information sources for practitioners looking for state-of-the art evidence to guide their decision-making and work practices ( Paré et al., 2015 ). Further, high-quality reviews become frequently cited pieces of work which researchers seek out as a first clear outline of the literature when undertaking empirical studies ( Cooper, 1988 ; Rowe, 2014 ). Scholars who track and gauge the impact of articles have found that review papers are cited and downloaded more often than any other type of published article ( Cronin, Ryan, & Coughlan, 2008 ; Montori, Wilczynski, Morgan, Haynes, & Hedges, 2003 ; Patsopoulos, Analatos, & Ioannidis, 2005 ). The reason for their popularity may be the fact that reading the review enables one to have an overview, if not a detailed knowledge of the area in question, as well as references to the most useful primary sources ( Cronin et al., 2008 ). Although they are not easy to conduct, the commitment to complete a review article provides a tremendous service to one’s academic community ( Paré et al., 2015 ; Petticrew & Roberts, 2006 ). Most, if not all, peer-reviewed journals in the fields of medical informatics publish review articles of some type.

The main objectives of this chapter are fourfold: (a) to provide an overview of the major steps and activities involved in conducting a stand-alone literature review; (b) to describe and contrast the different types of review articles that can contribute to the eHealth knowledge base; (c) to illustrate each review type with one or two examples from the eHealth literature; and (d) to provide a series of recommendations for prospective authors of review articles in this domain.

9.2. Overview of the Literature Review Process and Steps

As explained in Templier and Paré (2015) , there are six generic steps involved in conducting a review article:

• formulating the research question(s) and objective(s),
• searching the extant literature,
• screening for inclusion,
• assessing the quality of primary studies,
• extracting data, and
• analyzing data.

Although these steps are presented here in sequential order, one must keep in mind that the review process can be iterative and that many activities can be initiated during the planning stage and later refined during subsequent phases ( Finfgeld-Connett & Johnson, 2013 ; Kitchenham & Charters, 2007 ).

Formulating the research question(s) and objective(s): As a first step, members of the review team must appropriately justify the need for the review itself ( Petticrew & Roberts, 2006 ), identify the review’s main objective(s) ( Okoli & Schabram, 2010 ), and define the concepts or variables at the heart of their synthesis ( Cooper & Hedges, 2009 ; Webster & Watson, 2002 ). Importantly, they also need to articulate the research question(s) they propose to investigate ( Kitchenham & Charters, 2007 ). In this regard, we concur with Jesson, Matheson, and Lacey (2011) that clearly articulated research questions are key ingredients that guide the entire review methodology; they underscore the type of information that is needed, inform the search for and selection of relevant literature, and guide or orient the subsequent analysis. Searching the extant literature: The next step consists of searching the literature and making decisions about the suitability of material to be considered in the review ( Cooper, 1988 ). There exist three main coverage strategies. First, exhaustive coverage means an effort is made to be as comprehensive as possible in order to ensure that all relevant studies, published and unpublished, are included in the review and, thus, conclusions are based on this all-inclusive knowledge base. The second type of coverage consists of presenting materials that are representative of most other works in a given field or area. Often authors who adopt this strategy will search for relevant articles in a small number of top-tier journals in a field ( Paré et al., 2015 ). In the third strategy, the review team concentrates on prior works that have been central or pivotal to a particular topic. This may include empirical studies or conceptual papers that initiated a line of investigation, changed how problems or questions were framed, introduced new methods or concepts, or engendered important debate ( Cooper, 1988 ). Screening for inclusion: The following step consists of evaluating the applicability of the material identified in the preceding step ( Levy & Ellis, 2006 ; vom Brocke et al., 2009 ). Once a group of potential studies has been identified, members of the review team must screen them to determine their relevance ( Petticrew & Roberts, 2006 ). A set of predetermined rules provides a basis for including or excluding certain studies. This exercise requires a significant investment on the part of researchers, who must ensure enhanced objectivity and avoid biases or mistakes. As discussed later in this chapter, for certain types of reviews there must be at least two independent reviewers involved in the screening process and a procedure to resolve disagreements must also be in place ( Liberati et al., 2009 ; Shea et al., 2009 ). Assessing the quality of primary studies: In addition to screening material for inclusion, members of the review team may need to assess the scientific quality of the selected studies, that is, appraise the rigour of the research design and methods. Such formal assessment, which is usually conducted independently by at least two coders, helps members of the review team refine which studies to include in the final sample, determine whether or not the differences in quality may affect their conclusions, or guide how they analyze the data and interpret the findings ( Petticrew & Roberts, 2006 ). Ascribing quality scores to each primary study or considering through domain-based evaluations which study components have or have not been designed and executed appropriately makes it possible to reflect on the extent to which the selected study addresses possible biases and maximizes validity ( Shea et al., 2009 ). Extracting data: The following step involves gathering or extracting applicable information from each primary study included in the sample and deciding what is relevant to the problem of interest ( Cooper & Hedges, 2009 ). Indeed, the type of data that should be recorded mainly depends on the initial research questions ( Okoli & Schabram, 2010 ). However, important information may also be gathered about how, when, where and by whom the primary study was conducted, the research design and methods, or qualitative/quantitative results ( Cooper & Hedges, 2009 ). Analyzing and synthesizing data : As a final step, members of the review team must collate, summarize, aggregate, organize, and compare the evidence extracted from the included studies. The extracted data must be presented in a meaningful way that suggests a new contribution to the extant literature ( Jesson et al., 2011 ). Webster and Watson (2002) warn researchers that literature reviews should be much more than lists of papers and should provide a coherent lens to make sense of extant knowledge on a given topic. There exist several methods and techniques for synthesizing quantitative (e.g., frequency analysis, meta-analysis) and qualitative (e.g., grounded theory, narrative analysis, meta-ethnography) evidence ( Dixon-Woods, Agarwal, Jones, Young, & Sutton, 2005 ; Thomas & Harden, 2008 ).

9.3. Types of Review Articles and Brief Illustrations

EHealth researchers have at their disposal a number of approaches and methods for making sense out of existing literature, all with the purpose of casting current research findings into historical contexts or explaining contradictions that might exist among a set of primary research studies conducted on a particular topic. Our classification scheme is largely inspired from Paré and colleagues’ (2015) typology. Below we present and illustrate those review types that we feel are central to the growth and development of the eHealth domain.

9.3.1. Narrative Reviews

The narrative review is the “traditional” way of reviewing the extant literature and is skewed towards a qualitative interpretation of prior knowledge ( Sylvester et al., 2013 ). Put simply, a narrative review attempts to summarize or synthesize what has been written on a particular topic but does not seek generalization or cumulative knowledge from what is reviewed ( Davies, 2000 ; Green et al., 2006 ). Instead, the review team often undertakes the task of accumulating and synthesizing the literature to demonstrate the value of a particular point of view ( Baumeister & Leary, 1997 ). As such, reviewers may selectively ignore or limit the attention paid to certain studies in order to make a point. In this rather unsystematic approach, the selection of information from primary articles is subjective, lacks explicit criteria for inclusion and can lead to biased interpretations or inferences ( Green et al., 2006 ). There are several narrative reviews in the particular eHealth domain, as in all fields, which follow such an unstructured approach ( Silva et al., 2015 ; Paul et al., 2015 ).

Despite these criticisms, this type of review can be very useful in gathering together a volume of literature in a specific subject area and synthesizing it. As mentioned above, its primary purpose is to provide the reader with a comprehensive background for understanding current knowledge and highlighting the significance of new research ( Cronin et al., 2008 ). Faculty like to use narrative reviews in the classroom because they are often more up to date than textbooks, provide a single source for students to reference, and expose students to peer-reviewed literature ( Green et al., 2006 ). For researchers, narrative reviews can inspire research ideas by identifying gaps or inconsistencies in a body of knowledge, thus helping researchers to determine research questions or formulate hypotheses. Importantly, narrative reviews can also be used as educational articles to bring practitioners up to date with certain topics of issues ( Green et al., 2006 ).

Recently, there have been several efforts to introduce more rigour in narrative reviews that will elucidate common pitfalls and bring changes into their publication standards. Information systems researchers, among others, have contributed to advancing knowledge on how to structure a “traditional” review. For instance, Levy and Ellis (2006) proposed a generic framework for conducting such reviews. Their model follows the systematic data processing approach comprised of three steps, namely: (a) literature search and screening; (b) data extraction and analysis; and (c) writing the literature review. They provide detailed and very helpful instructions on how to conduct each step of the review process. As another methodological contribution, vom Brocke et al. (2009) offered a series of guidelines for conducting literature reviews, with a particular focus on how to search and extract the relevant body of knowledge. Last, Bandara, Miskon, and Fielt (2011) proposed a structured, predefined and tool-supported method to identify primary studies within a feasible scope, extract relevant content from identified articles, synthesize and analyze the findings, and effectively write and present the results of the literature review. We highly recommend that prospective authors of narrative reviews consult these useful sources before embarking on their work.

Darlow and Wen (2015) provide a good example of a highly structured narrative review in the eHealth field. These authors synthesized published articles that describe the development process of mobile health ( m-health ) interventions for patients’ cancer care self-management. As in most narrative reviews, the scope of the research questions being investigated is broad: (a) how development of these systems are carried out; (b) which methods are used to investigate these systems; and (c) what conclusions can be drawn as a result of the development of these systems. To provide clear answers to these questions, a literature search was conducted on six electronic databases and Google Scholar . The search was performed using several terms and free text words, combining them in an appropriate manner. Four inclusion and three exclusion criteria were utilized during the screening process. Both authors independently reviewed each of the identified articles to determine eligibility and extract study information. A flow diagram shows the number of studies identified, screened, and included or excluded at each stage of study selection. In terms of contributions, this review provides a series of practical recommendations for m-health intervention development.

9.3.2. Descriptive or Mapping Reviews

The primary goal of a descriptive review is to determine the extent to which a body of knowledge in a particular research topic reveals any interpretable pattern or trend with respect to pre-existing propositions, theories, methodologies or findings ( King & He, 2005 ; Paré et al., 2015 ). In contrast with narrative reviews, descriptive reviews follow a systematic and transparent procedure, including searching, screening and classifying studies ( Petersen, Vakkalanka, & Kuzniarz, 2015 ). Indeed, structured search methods are used to form a representative sample of a larger group of published works ( Paré et al., 2015 ). Further, authors of descriptive reviews extract from each study certain characteristics of interest, such as publication year, research methods, data collection techniques, and direction or strength of research outcomes (e.g., positive, negative, or non-significant) in the form of frequency analysis to produce quantitative results ( Sylvester et al., 2013 ). In essence, each study included in a descriptive review is treated as the unit of analysis and the published literature as a whole provides a database from which the authors attempt to identify any interpretable trends or draw overall conclusions about the merits of existing conceptualizations, propositions, methods or findings ( Paré et al., 2015 ). In doing so, a descriptive review may claim that its findings represent the state of the art in a particular domain ( King & He, 2005 ).

In the fields of health sciences and medical informatics, reviews that focus on examining the range, nature and evolution of a topic area are described by Anderson, Allen, Peckham, and Goodwin (2008) as mapping reviews . Like descriptive reviews, the research questions are generic and usually relate to publication patterns and trends. There is no preconceived plan to systematically review all of the literature although this can be done. Instead, researchers often present studies that are representative of most works published in a particular area and they consider a specific time frame to be mapped.

An example of this approach in the eHealth domain is offered by DeShazo, Lavallie, and Wolf (2009). The purpose of this descriptive or mapping review was to characterize publication trends in the medical informatics literature over a 20-year period (1987 to 2006). To achieve this ambitious objective, the authors performed a bibliometric analysis of medical informatics citations indexed in medline using publication trends, journal frequencies, impact factors, Medical Subject Headings (MeSH) term frequencies, and characteristics of citations. Findings revealed that there were over 77,000 medical informatics articles published during the covered period in numerous journals and that the average annual growth rate was 12%. The MeSH term analysis also suggested a strong interdisciplinary trend. Finally, average impact scores increased over time with two notable growth periods. Overall, patterns in research outputs that seem to characterize the historic trends and current components of the field of medical informatics suggest it may be a maturing discipline (DeShazo et al., 2009).

9.3.3. Scoping Reviews

Scoping reviews attempt to provide an initial indication of the potential size and nature of the extant literature on an emergent topic (Arksey & O’Malley, 2005; Daudt, van Mossel, & Scott, 2013 ; Levac, Colquhoun, & O’Brien, 2010). A scoping review may be conducted to examine the extent, range and nature of research activities in a particular area, determine the value of undertaking a full systematic review (discussed next), or identify research gaps in the extant literature ( Paré et al., 2015 ). In line with their main objective, scoping reviews usually conclude with the presentation of a detailed research agenda for future works along with potential implications for both practice and research.

Unlike narrative and descriptive reviews, the whole point of scoping the field is to be as comprehensive as possible, including grey literature (Arksey & O’Malley, 2005). Inclusion and exclusion criteria must be established to help researchers eliminate studies that are not aligned with the research questions. It is also recommended that at least two independent coders review abstracts yielded from the search strategy and then the full articles for study selection ( Daudt et al., 2013 ). The synthesized evidence from content or thematic analysis is relatively easy to present in tabular form (Arksey & O’Malley, 2005; Thomas & Harden, 2008 ).

One of the most highly cited scoping reviews in the eHealth domain was published by Archer, Fevrier-Thomas, Lokker, McKibbon, and Straus (2011) . These authors reviewed the existing literature on personal health record ( phr ) systems including design, functionality, implementation, applications, outcomes, and benefits. Seven databases were searched from 1985 to March 2010. Several search terms relating to phr s were used during this process. Two authors independently screened titles and abstracts to determine inclusion status. A second screen of full-text articles, again by two independent members of the research team, ensured that the studies described phr s. All in all, 130 articles met the criteria and their data were extracted manually into a database. The authors concluded that although there is a large amount of survey, observational, cohort/panel, and anecdotal evidence of phr benefits and satisfaction for patients, more research is needed to evaluate the results of phr implementations. Their in-depth analysis of the literature signalled that there is little solid evidence from randomized controlled trials or other studies through the use of phr s. Hence, they suggested that more research is needed that addresses the current lack of understanding of optimal functionality and usability of these systems, and how they can play a beneficial role in supporting patient self-management ( Archer et al., 2011 ).

9.3.4. Forms of Aggregative Reviews

Healthcare providers, practitioners, and policy-makers are nowadays overwhelmed with large volumes of information, including research-based evidence from numerous clinical trials and evaluation studies, assessing the effectiveness of health information technologies and interventions ( Ammenwerth & de Keizer, 2004 ; Deshazo et al., 2009 ). It is unrealistic to expect that all these disparate actors will have the time, skills, and necessary resources to identify the available evidence in the area of their expertise and consider it when making decisions. Systematic reviews that involve the rigorous application of scientific strategies aimed at limiting subjectivity and bias (i.e., systematic and random errors) can respond to this challenge.

Systematic reviews attempt to aggregate, appraise, and synthesize in a single source all empirical evidence that meet a set of previously specified eligibility criteria in order to answer a clearly formulated and often narrow research question on a particular topic of interest to support evidence-based practice ( Liberati et al., 2009 ). They adhere closely to explicit scientific principles ( Liberati et al., 2009 ) and rigorous methodological guidelines (Higgins & Green, 2008) aimed at reducing random and systematic errors that can lead to deviations from the truth in results or inferences. The use of explicit methods allows systematic reviews to aggregate a large body of research evidence, assess whether effects or relationships are in the same direction and of the same general magnitude, explain possible inconsistencies between study results, and determine the strength of the overall evidence for every outcome of interest based on the quality of included studies and the general consistency among them ( Cook, Mulrow, & Haynes, 1997 ). The main procedures of a systematic review involve:

• Formulating a review question and developing a search strategy based on explicit inclusion criteria for the identification of eligible studies (usually described in the context of a detailed review protocol).
• Searching for eligible studies using multiple databases and information sources, including grey literature sources, without any language restrictions.
• Selecting studies, extracting data, and assessing risk of bias in a duplicate manner using two independent reviewers to avoid random or systematic errors in the process.
• Analyzing data using quantitative or qualitative methods.
• Presenting results in summary of findings tables.
• Interpreting results and drawing conclusions.

Many systematic reviews, but not all, use statistical methods to combine the results of independent studies into a single quantitative estimate or summary effect size. Known as meta-analyses , these reviews use specific data extraction and statistical techniques (e.g., network, frequentist, or Bayesian meta-analyses) to calculate from each study by outcome of interest an effect size along with a confidence interval that reflects the degree of uncertainty behind the point estimate of effect ( Borenstein, Hedges, Higgins, & Rothstein, 2009 ; Deeks, Higgins, & Altman, 2008 ). Subsequently, they use fixed or random-effects analysis models to combine the results of the included studies, assess statistical heterogeneity, and calculate a weighted average of the effect estimates from the different studies, taking into account their sample sizes. The summary effect size is a value that reflects the average magnitude of the intervention effect for a particular outcome of interest or, more generally, the strength of a relationship between two variables across all studies included in the systematic review. By statistically combining data from multiple studies, meta-analyses can create more precise and reliable estimates of intervention effects than those derived from individual studies alone, when these are examined independently as discrete sources of information.

The review by Gurol-Urganci, de Jongh, Vodopivec-Jamsek, Atun, and Car (2013) on the effects of mobile phone messaging reminders for attendance at healthcare appointments is an illustrative example of a high-quality systematic review with meta-analysis. Missed appointments are a major cause of inefficiency in healthcare delivery with substantial monetary costs to health systems. These authors sought to assess whether mobile phone-based appointment reminders delivered through Short Message Service ( sms ) or Multimedia Messaging Service ( mms ) are effective in improving rates of patient attendance and reducing overall costs. To this end, they conducted a comprehensive search on multiple databases using highly sensitive search strategies without language or publication-type restrictions to identify all rct s that are eligible for inclusion. In order to minimize the risk of omitting eligible studies not captured by the original search, they supplemented all electronic searches with manual screening of trial registers and references contained in the included studies. Study selection, data extraction, and risk of bias assessments were performed inde­­pen­dently by two coders using standardized methods to ensure consistency and to eliminate potential errors. Findings from eight rct s involving 6,615 participants were pooled into meta-analyses to calculate the magnitude of effects that mobile text message reminders have on the rate of attendance at healthcare appointments compared to no reminders and phone call reminders.

Meta-analyses are regarded as powerful tools for deriving meaningful conclusions. However, there are situations in which it is neither reasonable nor appropriate to pool studies together using meta-analytic methods simply because there is extensive clinical heterogeneity between the included studies or variation in measurement tools, comparisons, or outcomes of interest. In these cases, systematic reviews can use qualitative synthesis methods such as vote counting, content analysis, classification schemes and tabulations, as an alternative approach to narratively synthesize the results of the independent studies included in the review. This form of review is known as qualitative systematic review.

A rigorous example of one such review in the eHealth domain is presented by Mickan, Atherton, Roberts, Heneghan, and Tilson (2014) on the use of handheld computers by healthcare professionals and their impact on access to information and clinical decision-making. In line with the methodological guide­lines for systematic reviews, these authors: (a) developed and registered with prospero ( www.crd.york.ac.uk/ prospero / ) an a priori review protocol; (b) conducted comprehensive searches for eligible studies using multiple databases and other supplementary strategies (e.g., forward searches); and (c) subsequently carried out study selection, data extraction, and risk of bias assessments in a duplicate manner to eliminate potential errors in the review process. Heterogeneity between the included studies in terms of reported outcomes and measures precluded the use of meta-analytic methods. To this end, the authors resorted to using narrative analysis and synthesis to describe the effectiveness of handheld computers on accessing information for clinical knowledge, adherence to safety and clinical quality guidelines, and diagnostic decision-making.

In recent years, the number of systematic reviews in the field of health informatics has increased considerably. Systematic reviews with discordant findings can cause great confusion and make it difficult for decision-makers to interpret the review-level evidence ( Moher, 2013 ). Therefore, there is a growing need for appraisal and synthesis of prior systematic reviews to ensure that decision-making is constantly informed by the best available accumulated evidence. Umbrella reviews , also known as overviews of systematic reviews, are tertiary types of evidence synthesis that aim to accomplish this; that is, they aim to compare and contrast findings from multiple systematic reviews and meta-analyses ( Becker & Oxman, 2008 ). Umbrella reviews generally adhere to the same principles and rigorous methodological guidelines used in systematic reviews. However, the unit of analysis in umbrella reviews is the systematic review rather than the primary study ( Becker & Oxman, 2008 ). Unlike systematic reviews that have a narrow focus of inquiry, umbrella reviews focus on broader research topics for which there are several potential interventions ( Smith, Devane, Begley, & Clarke, 2011 ). A recent umbrella review on the effects of home telemonitoring interventions for patients with heart failure critically appraised, compared, and synthesized evidence from 15 systematic reviews to investigate which types of home telemonitoring technologies and forms of interventions are more effective in reducing mortality and hospital admissions ( Kitsiou, Paré, & Jaana, 2015 ).

9.3.5. Realist Reviews

Realist reviews are theory-driven interpretative reviews developed to inform, enhance, or supplement conventional systematic reviews by making sense of heterogeneous evidence about complex interventions applied in diverse contexts in a way that informs policy decision-making ( Greenhalgh, Wong, Westhorp, & Pawson, 2011 ). They originated from criticisms of positivist systematic reviews which centre on their “simplistic” underlying assumptions ( Oates, 2011 ). As explained above, systematic reviews seek to identify causation. Such logic is appropriate for fields like medicine and education where findings of randomized controlled trials can be aggregated to see whether a new treatment or intervention does improve outcomes. However, many argue that it is not possible to establish such direct causal links between interventions and outcomes in fields such as social policy, management, and information systems where for any intervention there is unlikely to be a regular or consistent outcome ( Oates, 2011 ; Pawson, 2006 ; Rousseau, Manning, & Denyer, 2008 ).

To circumvent these limitations, Pawson, Greenhalgh, Harvey, and Walshe (2005) have proposed a new approach for synthesizing knowledge that seeks to unpack the mechanism of how “complex interventions” work in particular contexts. The basic research question — what works? — which is usually associated with systematic reviews changes to: what is it about this intervention that works, for whom, in what circumstances, in what respects and why? Realist reviews have no particular preference for either quantitative or qualitative evidence. As a theory-building approach, a realist review usually starts by articulating likely underlying mechanisms and then scrutinizes available evidence to find out whether and where these mechanisms are applicable ( Shepperd et al., 2009 ). Primary studies found in the extant literature are viewed as case studies which can test and modify the initial theories ( Rousseau et al., 2008 ).

The main objective pursued in the realist review conducted by Otte-Trojel, de Bont, Rundall, and van de Klundert (2014) was to examine how patient portals contribute to health service delivery and patient outcomes. The specific goals were to investigate how outcomes are produced and, most importantly, how variations in outcomes can be explained. The research team started with an exploratory review of background documents and research studies to identify ways in which patient portals may contribute to health service delivery and patient outcomes. The authors identified six main ways which represent “educated guesses” to be tested against the data in the evaluation studies. These studies were identified through a formal and systematic search in four databases between 2003 and 2013. Two members of the research team selected the articles using a pre-established list of inclusion and exclusion criteria and following a two-step procedure. The authors then extracted data from the selected articles and created several tables, one for each outcome category. They organized information to bring forward those mechanisms where patient portals contribute to outcomes and the variation in outcomes across different contexts.

9.3.6. Critical Reviews

Lastly, critical reviews aim to provide a critical evaluation and interpretive analysis of existing literature on a particular topic of interest to reveal strengths, weaknesses, contradictions, controversies, inconsistencies, and/or other important issues with respect to theories, hypotheses, research methods or results ( Baumeister & Leary, 1997 ; Kirkevold, 1997 ). Unlike other review types, critical reviews attempt to take a reflective account of the research that has been done in a particular area of interest, and assess its credibility by using appraisal instruments or critical interpretive methods. In this way, critical reviews attempt to constructively inform other scholars about the weaknesses of prior research and strengthen knowledge development by giving focus and direction to studies for further improvement ( Kirkevold, 1997 ).

Kitsiou, Paré, and Jaana (2013) provide an example of a critical review that assessed the methodological quality of prior systematic reviews of home telemonitoring studies for chronic patients. The authors conducted a comprehensive search on multiple databases to identify eligible reviews and subsequently used a validated instrument to conduct an in-depth quality appraisal. Results indicate that the majority of systematic reviews in this particular area suffer from important methodological flaws and biases that impair their internal validity and limit their usefulness for clinical and decision-making purposes. To this end, they provide a number of recommendations to strengthen knowledge development towards improving the design and execution of future reviews on home telemonitoring.

9.4. Summary

Table 9.1 outlines the main types of literature reviews that were described in the previous sub-sections and summarizes the main characteristics that distinguish one review type from another. It also includes key references to methodological guidelines and useful sources that can be used by eHealth scholars and researchers for planning and developing reviews.

Typology of Literature Reviews (adapted from Paré et al., 2015).

As shown in Table 9.1 , each review type addresses different kinds of research questions or objectives, which subsequently define and dictate the methods and approaches that need to be used to achieve the overarching goal(s) of the review. For example, in the case of narrative reviews, there is greater flexibility in searching and synthesizing articles ( Green et al., 2006 ). Researchers are often relatively free to use a diversity of approaches to search, identify, and select relevant scientific articles, describe their operational characteristics, present how the individual studies fit together, and formulate conclusions. On the other hand, systematic reviews are characterized by their high level of systematicity, rigour, and use of explicit methods, based on an “a priori” review plan that aims to minimize bias in the analysis and synthesis process (Higgins & Green, 2008). Some reviews are exploratory in nature (e.g., scoping/mapping reviews), whereas others may be conducted to discover patterns (e.g., descriptive reviews) or involve a synthesis approach that may include the critical analysis of prior research ( Paré et al., 2015 ). Hence, in order to select the most appropriate type of review, it is critical to know before embarking on a review project, why the research synthesis is conducted and what type of methods are best aligned with the pursued goals.

9.5. Concluding Remarks

In light of the increased use of evidence-based practice and research generating stronger evidence ( Grady et al., 2011 ; Lyden et al., 2013 ), review articles have become essential tools for summarizing, synthesizing, integrating or critically appraising prior knowledge in the eHealth field. As mentioned earlier, when rigorously conducted review articles represent powerful information sources for eHealth scholars and practitioners looking for state-of-the-art evidence. The typology of literature reviews we used herein will allow eHealth researchers, graduate students and practitioners to gain a better understanding of the similarities and differences between review types.

We must stress that this classification scheme does not privilege any specific type of review as being of higher quality than another ( Paré et al., 2015 ). As explained above, each type of review has its own strengths and limitations. Having said that, we realize that the methodological rigour of any review — be it qualitative, quantitative or mixed — is a critical aspect that should be considered seriously by prospective authors. In the present context, the notion of rigour refers to the reliability and validity of the review process described in section 9.2. For one thing, reliability is related to the reproducibility of the review process and steps, which is facilitated by a comprehensive documentation of the literature search process, extraction, coding and analysis performed in the review. Whether the search is comprehensive or not, whether it involves a methodical approach for data extraction and synthesis or not, it is important that the review documents in an explicit and transparent manner the steps and approach that were used in the process of its development. Next, validity characterizes the degree to which the review process was conducted appropriately. It goes beyond documentation and reflects decisions related to the selection of the sources, the search terms used, the period of time covered, the articles selected in the search, and the application of backward and forward searches ( vom Brocke et al., 2009 ). In short, the rigour of any review article is reflected by the explicitness of its methods (i.e., transparency) and the soundness of the approach used. We refer those interested in the concepts of rigour and quality to the work of Templier and Paré (2015) which offers a detailed set of methodological guidelines for conducting and evaluating various types of review articles.

To conclude, our main objective in this chapter was to demystify the various types of literature reviews that are central to the continuous development of the eHealth field. It is our hope that our descriptive account will serve as a valuable source for those conducting, evaluating or using reviews in this important and growing domain.

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• Cite this Page Paré G, Kitsiou S. Chapter 9 Methods for Literature Reviews. In: Lau F, Kuziemsky C, editors. Handbook of eHealth Evaluation: An Evidence-based Approach [Internet]. Victoria (BC): University of Victoria; 2017 Feb 27.
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CASE REPORT article

Ureter mixed neuroendocrine-non-neuroendocrine neoplasm: a case report and literature review.

• 1 Department of Pathology, Second Affiliated Hospital of Jiujiang University, Jiujiang, China
• 2 Department of Clinical Laboratory, Wuhan Jinyintan Hospital, Wuhan, China
• 3 Department of General Surgery, Second Affiliated Hospital of Jiujiang University, Jiujiang, China
• 4 Department of Pathology, Jiujiang First People’s Hospital, Jiujiang, China
• 5 Department of Pathology, Yangpu Hospital, School of Medicine, Tongji University, Shanghai, China

Cases of mixed neuroendocrine-non-neuroendocrine neoplasms (MiNENs) of the urinary system are rare, and reports of primary MiNENs in the ureter are lacking. Herein, we present the case of a 71-year-old man who presented with painless gross hematuria and weight loss. Contrast-enhanced abdominal computed tomography (CT) revealed a tumor, comprising small cell neuroendocrine carcinoma (SCNEC) and adenocarcinomatous components, attached to the ureter. The SCNEC components were strongly positive for synaptophysin, CD56 and INSM1 and adenocarcinomatous components were strongly positive for CDX2 and cytokeratin 20, respectively. Four weeks post-surgery, the patient received four cycles of cisplatin-based chemotherapy; the 7-month follow-up CT confirmed that he was healthy without disease recurrence. The occurrence of MiNEN in the ureter with SCNEC and adenocarcinomatous components is extremely rare, wherein histopathological and immunohistochemical features aid in the diagnosis MiNEN. With its aggressive nature, MiNEN can only be effectively treated by early diagnosis and radical surgery.

Introduction

Mixed neuroendocrine-non-neuroendocrine neoplasms (MiNENs) are characterized by the presence of two major neuroendocrine and non-neuroendocrine morphological components, each accounting for >30% of the total lesion ( 1 ). MiNENs have been commonly detected in the gastrointestinal tract and recently in many other organs including the cervix, bladder, and tongue ( 2 , 3 ). Urothelial carcinoma represents >90% of all malignancies in the urinary system. However, MiNEN that originate in the ureter, including neuroendocrine carcinomas (NECs) and urothelial carcinomas, are rare ( 4 ). Although the developmental mechanism closely relates to the genetic events in tumor cells, the biological behavior, prognosis, and molecular origin of MiNENs remain unclear.

Here, we report a case of a patient with a primary MiNEN of the ureter that comprised small cell neuroendocrine carcinoma (SCNEC) and adenocarcinomatous components. Additionally, we have summarized the relevant literature, to improve the medical community’s understanding of this rare type of tumor.

Case description

A 71-year-old man presented with a 3-month history of painless gross hematuria and a 2-month history of weight loss. The patient had a 32-year and 43-year history of smoking and alcohol abuse, respectively. The patient was previously fit and had no specific life history. The physical examination findings were unremarkable. Laboratory investigations revealed that the routine blood, electrolytes, liver function, and serum amylase levels were within the reference ranges, urinalysis revealed red blood cells (3+), and negative levels of the serum tumor markers PSA, BTA and AFP. The urine cytological analysis revealed malignant epithelial neoplastic cells. Computed tomography (CT) revealed a 25-mm extensive heterogeneously-enhanced mass in the middle and lower part of his left ureteral segment and left hydronephrosis ( Figure 1A ). A subsequent ureteroscopic biopsy revealed poorly differentiated tumors. Positron emission tomography-CT and gastrointestinal endoscopy confirmed the lack of primary or metastatic lesions in other organs. The patient underwent laparoscopic left nephroureterectomy with bladder cuff resection and retroperitoneal lymph node removal. The patient has been followed up with CT postoperatively without ultrasound. Cystoscopy was normal and PET CT showed no other lesions. Postoperative laboratory indicators: all indicators are still normal, urine routine and test hematuria returned to normal. Four weeks post-discharge, the patient received four cycles of cisplatin and etoposide chemotherapy. At his 7-month follow-up, no recurrence or metastasis was noted; follow-up care is continued.

Figure 1 (A) Contrast-enhanced computerized tomography (CECT) of the left ureteral segment and left hydronephrosis. CECT revealed a 20-mm lesion on the left ureteral segment. Neuroendocrine carcinoma and adenocarcinoma in biopsy. Low ( (B) , hematoxylin-eosin, × 40) and intermediate [ (C) , hematoxylin-eosin, × 200] magnification showing adenocarcinoma and Neuroendocrine carcinoma. Neuroendocrine in biopsy: High magnification [ (D) , hematoxylin-eosin, × 400]. Adenocarcinoma in biopsy: High magnification [ (E) , hematoxylin-eosin, × 400].

The immunohistochemical study was performed on 3-μm-thick sections using prediluted ready-to-use vials of the antibodies with an automated immunostainer.

The tissue sent for examination was a specimen of the left kidney, ureter, and part of the bladder. The ureter was 26 cm in length, and 9 cm from the opening of the ureter, a 2.5 cm × 2 cm ×1 cm mass was visible that was grayish-white in color on the section, brittle, with unclear borders, and without necrosis. Microscopically, the tumor protruding into the lumen comprised two intimately admixed morphological tumor cells ( Figures 1B, C ). Most cells (55%) exhibited solid and trabecular growth, were small, and had round or spindle-shapes, scant cytoplasm, dark staining, inconspicuous nucleoli, extensive necrosis, and a mitotic index of 60/10 high-power fields (HPFs) ( Figures 1D ). The other 45% of tumor cells exhibited glandular lumens and papillary formation, with larger tumor cells, columnar and cubic, abundant cytoplasm, conspicuous nucleoli, enteric and mucinous differentiation, and a mitotic index of 4/10 HPFs ( Figures 1E ). Conventional urothelial carcinoma components were not detected, and the tumor invaded the deep muscle. Lymph node dissection information: abdominal lymph nodes, common iliac lymph nodes, external iliac lymph nodes, obturator lymph nodes, and internal iliac lymph nodes, 0.2-1 cm in diameter. However, renal parenchymal, arteriovenous, periureteral tissue, and lymph node involvement were absent. The overall pathological stage at diagnosis was pT2N0M0.

Immunohistochemistry showed positive staining in different histological components ( Figures 2A–J ). The tumor cells in the solid components were positive for synaptophysin, CD56 and INSM1, partly positive for TTF-1 and negative for SSTR2. Staining with chromogranin A was very weak, and staining for CDX2, SATB2, and cytokeratin (CK)20 was negative. The tumor cells in the glandular components were positive for CDX2 and CK20 and focally positive for SATB2. The cells tested negative for synaptophysin, CD56, chromogranin A, and TTF1. Both tumor cell components were positive for CKpan, p53, MLH1, PMS2, MSH2, and MSH6 and were negative for GATA3, LCA, and HER2. The Ki-67 indices of tumor cells in the solid and glandular components were approximately 80% and 30%, respectively.

Figure 2 Neuroendocrine carcinoma and adenocarcinoma in biopsy. Low [ (A) , hematoxylin-eosin, × 40]. Immunohistochemical staining showed positivity for a urothelial marker [(GATA3 (B) ], neuroendocrine marker [synaptophysin (C) ], adenocarcinoma marker [CDX2 (D) ], a neuroendocrine marker [CD56 (E) ], adenocarcinoma marker [CDX2 (F) , epithelial marker (CKpan (G) ], a neuroendocrine marker [INSM1 (H) ], a neuroendocrine marker [SSTR2 (I) ] and proliferation index marker [Ki67 (J) ].

Finally, we diagnosed the patient with primary MiNEN of the ureter, with poorly differentiated neuroendocrine carcinoma (SCNEC, 55%) and moderately and highly differentiated non-neuroendocrine carcinoma (adenocarcinoma, 45%).

NECs of the urinary system include SCNECs, large cell neuroendocrine carcinoma (LCNEC), well-differentiated neuroendocrine tumors, and paragangliomas and account for <0.5% of all urothelial neoplasms. Over half of the primary urinary NECs are mixed with other histological components ( 5 , 6 ), and more than 90% of these reports of non-neuroendocrine components were of urothelial carcinoma, followed by squamous cell carcinoma, lymphoma, sarcoma, and adenocarcinoma, in order of frequency. We summarized published cases of mixed neuroendocrine and adenocarcinomatous tumor of the urinary system between 1990 and 2023 ( Table 1 ) ( 7 – 13 ). The male-to-female ratio was 7:1, with a median of 61 years, and it can occur in the bladder, ureter and urethra. Specifically, SCNEC is the most common NEC type, while the non-neuroendocrine components can be simple adenocarcinoma or mixed with other components. Unfortunately, the proportion of components has not been reported in most cases. In World Health Organization (WHO) classification 5th edition (2022) of urogenital tumors, mixed neuroendocrine neoplasms are still not defined as MiNEN with 30% component criteria in the manner of gastrointestinal tumors, possibly because of evidence that only a small portion of neuroendocrine carcinoma has a prognostic impact in urinary tract tumors ( 14 ). Nevertheless, in order to better understanding of this rare case of Ureter MiNEN, especially when they are morphological high-grade, more detailed clinicopathological data and percentages of each component are necessary, because they still may influence prognosis.

Table 1 Published cases of mixed neuroendocrine and adenocarcinomatous tumor of the urinary system.

Xu et al. ( 15 ) evaluated 17 patients and reported that ureteral collision carcinoma, especially those occurring in the lower right ureter, were common in older Asian men. Concordantly, Zhong et al. ( 16 ) evaluated 32 patients with ureteral SCNECs and suggested that the cancers may be related to genetic, environmental, and lifestyle habits in Asians. This may be attributed to a higher incidence of smoking exposure, drug abuse (Chinese herbal medicine, painkillers), occupational exposure to certain aromatic amines, and lithiasis concomitant infections among Asians ( 4 , 17 ). Our patient was an older Asian man with a 32-year history of smoking, consistent with the literature reports; however, the lesion was in the middle and lower left ureter, unlike previous literature reports that it is more common in the lower right ureter.

The preoperative diagnosis of ureteral MiNEN is confounded by uncharacteristic symptoms, including painless gross hematuria and a ureter mass on imaging; additionally, the accuracy of preoperative biopsy remains controversial. Therefore, final confirmation requires complete surgical excision for examination ( 18 ). Combining the clinical history and imaging findings to exclude gastrointestinal and bladder metastases is crucial for the primary diagnosis. In our case, the histomorphological features were comprehensively evaluated, and the differential diagnosis was based on immunohistochemical positivity for synaptophysin, chromogranin A, CD56, neuron-specific enolase, and characteristic markers of the corresponding non-neuroendocrine components. Furthermore, urinary cytology and biopsy pathology revealed malignancy; therefore, we completely surgically resected the tumor. Combined with the medical history, negative imaging, and spectroscopic nodes to exclude metastasis, we diagnosed the patient with primary ureteral MiNEN.

The histogenesis of the primary ureteral MiNEN remains unclear, but has been discussed extensively. At present, many scholars support the theory of MiNEN is hypothesized to originate from monoclonal pluripotent stem cells that differentiate bidirectionally when stimulated with certain factors, which could explain the mixed histological features in urogenital NECs ( 19 ). Tränkenschuh et al. ( 11 ) reported the case of a patient with primary adenoneuroendocrine carcinoma of the bladder combined with in situ urothelial carcinoma and observed significant similarities in the molecular features between these carcinomas, thereby highlighting their common clonal origin. Another postulated theory is that ureteral MiNEN may be caused by conversion of non-neuroendocrine to neuroendocrine components during tumor progression, including originating from residual urachus epithelium, or cystitis glandularis and intestinal anisocytosis, which would seem to explain the mix of adenocarcinoma components in MiNEN. Eckstein et al. ( 20 ) described the case of a patient with adenoneuroendocrine carcinoma of the bladder, with significant cystitis glandularis and intestinal anisocytosis surrounding the tumor. The investigators suggested that this carcinoma developed from primary adenocarcinoma of the bladder. In our case, epithelial neuroendocrine cells transformation such as glandular or intestinal epithelial chemotaxis components in the mucosa surrounding the ureteral MiNEN were not detected. In fact, almost half of urogenital NECs patients had other tumor components such as UC, squamous cell carcinoma, adenocarcinoma. Meanwhile, the conversion of non-neuroendocrine components to neuroendocrine components is rarely reported, thus further buttresses the hypothesis of the origin of multipotent stem cells.

At present, the genetic characteristics of MiNENs containing adenocarcinoma components have been investigated in the digestive system, and it is suggested that CCNE1 gain and FAT1 loss might promote the tumorigenesis of MiNENs partially through regulating cell cycle G1/S checkpoint signaling. Moreover, loss of MAPK1 and alterations in the MAPK signaling pathway, specifically occurred in the NEC component, which might contribute to the neuroendocrine differentiation ( 21 ). However, due to its rare molecular changes in the urinary system have not been reported, the relevant studies of single components can provide some references. In a report about urinary molecular typing of bladder SCNEC, a significant proportion of these tumors expressed the c-kit, TP53 and RB1 gene mutations ( 22 , 23 ). Furthermore, whole exome sequencing of urinary system adenocarcinoma has found that prevalent T>A substitutions were observed among somatic mutations, and major trinucleotide contexts included 5’-CTC-3’ and 5’-CTG-3’ ( 24 ). Lu et al ( 25 ). Explore the genomic alterations of ureteral hepatoid adenocarcinoma, driver somatic mutations of TP53 and KMT2D genes were found, indicating that ureteral hepatoid adenocarcinoma has similar mutational characteristics to urothelial carcinoma. These genetic characteristics have certain potential significance for future targeted therapy and prognosis judgment, and need to be confirmed by further studies on large samples.

We possess limited treatment experience owing to the rarity of ureteral MiNEN; thus, nephroureterectomy with bladder cuff resection is preferred ( 4 , 15 ). Furthermore, postoperative platinum-based adjuvant chemotherapy for highly malignant SCNECs components significantly improves prognosis compared with surgical resection alone ( 5 , 26 ). Qing et al. ( 27 ) attempted to treat primary ureteral SCNECs with a combination of PD-L1 ICIs and radiotherapy. Despite the unsatisfactory outcome, the report provided new insights into various treatment strategies. Furthermore, the prognosis of MiNENs is driven by their high-grade neuroendocrine components, especially in the digestive system; therefore, their prognosis is comparable with that of pure neuroendocrine cancers ( 28 , 29 ). However, another large sample of urinary MiNENs was considered to be better than that of pure SCNECs ( 30 ). Pini et al. ( 9 ) reported that case of a patient with MiNEN (comprising LCNEC and adenocarcinoma) of the bladder and observed that mucinous adenocarcinoma was the most aggressive component. These findings suggest that non-neuroendocrine carcinomas are likely crucial in determining patient quality of life and prognosis. The prognostic factors of urinary MiNEN are closely related to the pathological stage; however, their association with sex remains controversial ( 5 , 30 ). This disparity may be related to the rarity of the case, diverse sample sources or limited case numbers. In future work, increasing case collection and pooled case reports can make prognosis clearer. Anyhow, ureter MiNENs have a poor prognosis, which have an overall survival of approximately 15 months and a 3-year survival rate of less than 30%, with metastases occurring within 13 months in most patients. Furthermore, the most common target organs are the lungs and brain ( 5 ). In our patient, we performed a laparoscopic left nephroureterectomy with bladder cuff resection, followed by cisplatin and etoposide chemotherapy. We observed no recurrence during the 7-month follow-up. This outcome may relate to the T2 pathological stage, increased proportion of well-differentiated adenocarcinoma components, and shorter follow-up time. And a timeline figure summarizing the case diagnosis and treatment pathway was shown in Figure 3 .

Figure 3 A timeline figure summarising the case diagnosis and treatment pathway.

In conclusion, we report a rare case of a patient with ureteral MiNEN with SCNECs and adenocarcinoma components, whose clinical imaging presentation was mostly atypical. Characteristic histological patterns and immunohistochemical markers facilitated the accurate diagnosis. Although we lack standard MiNEN treatment options owing to its rarity, radical ureteral resection and postoperative chemotherapy can help improve prognosis, as in the cases of ureteral small cell carcinoma and adenocarcinoma.

Data availability statement

The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.

Ethics statement

The studies involving humans were approved by ethics committee of Second Affiliated Hospital of Jiujiang University. The studies were conducted in accordance with the local legislation and institutional requirements. The participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.

Author contributions

BZ: Conceptualization, Data curation, Investigation, Visualization, Writing – original draft. XG: Conceptualization, Investigation, Writing – original draft. XL: Investigation, Visualization, Writing – original draft. LP: Methodology, Writing – original draft. HH: Conceptualization, Data curation, Formal Analysis, Funding acquisition, Investigation, Project administration, Supervision, Visualization, Writing – review & editing.

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This work was supported by Climb Plan of Tongji University Affiliated Yangpu Hospital (No.Ye2202105) and Project of College-level Key Discipline of Tongji University Affiliated Yangpu Hospital (No.2023YJXK01).

Acknowledgments

The authors would like to acknowledge the Department of Pathology of Jiujiang First People’s Hospital for their assistance with providing research materials and digitizing the slides in this research. We thank Editage ( www.editage.cn ) for its linguistic assistance during the preparation of this manuscript.

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.

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Keywords: ureter, mixed neuroendocrine-non-neuroendocrine neoplasm, MiNEN, immunohistochemistry, diagnosis

Citation: Zhou B, Gan X, Li X, Peng L and Hao H (2024) Ureter mixed neuroendocrine-non-neuroendocrine neoplasm: a case report and literature review. Front. Oncol. 14:1390350. doi: 10.3389/fonc.2024.1390350

Received: 23 February 2024; Accepted: 13 May 2024; Published: 31 May 2024.

Reviewed by:

Copyright © 2024 Zhou, Gan, Li, Peng and Hao. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY) . The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

*Correspondence: Hua Hao, [email protected]

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