what is peer review literature

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• v.59(1); 2021

Peer review guidance: a primer for researchers

Olena zimba.

1 Department of Internal Medicine No. 2, Danylo Halytsky Lviv National Medical University, Lviv, Ukraine

Armen Yuri Gasparyan

2 Departments of Rheumatology and Research and Development, Dudley Group NHS Foundation Trust (Teaching Trust of the University of Birmingham, UK), Russells Hall Hospital, Dudley, West Midlands, UK

The peer review process is essential for quality checks and validation of journal submissions. Although it has some limitations, including manipulations and biased and unfair evaluations, there is no other alternative to the system. Several peer review models are now practised, with public review being the most appropriate in view of the open science movement. Constructive reviewer comments are increasingly recognised as scholarly contributions which should meet certain ethics and reporting standards. The Publons platform, which is now part of the Web of Science Group (Clarivate Analytics), credits validated reviewer accomplishments and serves as an instrument for selecting and promoting the best reviewers. All authors with relevant profiles may act as reviewers. Adherence to research reporting standards and access to bibliographic databases are recommended to help reviewers draft evidence-based and detailed comments.

Introduction

The peer review process is essential for evaluating the quality of scholarly works, suggesting corrections, and learning from other authors’ mistakes. The principles of peer review are largely based on professionalism, eloquence, and collegiate attitude. As such, reviewing journal submissions is a privilege and responsibility for ‘elite’ research fellows who contribute to their professional societies and add value by voluntarily sharing their knowledge and experience.

Since the launch of the first academic periodicals back in 1665, the peer review has been mandatory for validating scientific facts, selecting influential works, and minimizing chances of publishing erroneous research reports [ 1 ]. Over the past centuries, peer review models have evolved from single-handed editorial evaluations to collegial discussions, with numerous strengths and inevitable limitations of each practised model [ 2 , 3 ]. With multiplication of periodicals and editorial management platforms, the reviewer pool has expanded and internationalized. Various sets of rules have been proposed to select skilled reviewers and employ globally acceptable tools and language styles [ 4 , 5 ].

In the era of digitization, the ethical dimension of the peer review has emerged, necessitating involvement of peers with full understanding of research and publication ethics to exclude unethical articles from the pool of evidence-based research and reviews [ 6 ]. In the time of the COVID-19 pandemic, some, if not most, journals face the unavailability of skilled reviewers, resulting in an unprecedented increase of articles without a history of peer review or those with surprisingly short evaluation timelines [ 7 ].

Editorial recommendations and the best reviewers

Guidance on peer review and selection of reviewers is currently available in the recommendations of global editorial associations which can be consulted by journal editors for updating their ethics statements and by research managers for crediting the evaluators. The International Committee on Medical Journal Editors (ICMJE) qualifies peer review as a continuation of the scientific process that should involve experts who are able to timely respond to reviewer invitations, submitting unbiased and constructive comments, and keeping confidentiality [ 8 ].

The reviewer roles and responsibilities are listed in the updated recommendations of the Council of Science Editors (CSE) [ 9 ] where ethical conduct is viewed as a premise of the quality evaluations. The Committee on Publication Ethics (COPE) further emphasizes editorial strategies that ensure transparent and unbiased reviewer evaluations by trained professionals [ 10 ]. Finally, the World Association of Medical Editors (WAME) prioritizes selecting the best reviewers with validated profiles to avoid substandard or fraudulent reviewer comments [ 11 ]. Accordingly, the Sarajevo Declaration on Integrity and Visibility of Scholarly Publications encourages reviewers to register with the Open Researcher and Contributor ID (ORCID) platform to validate and publicize their scholarly activities [ 12 ].

Although the best reviewer criteria are not listed in the editorial recommendations, it is apparent that the manuscript evaluators should be active researchers with extensive experience in the subject matter and an impressive list of relevant and recent publications [ 13 ]. All authors embarking on an academic career and publishing articles with active contact details can be involved in the evaluation of others’ scholarly works [ 14 ]. Ideally, the reviewers should be peers of the manuscript authors with equal scholarly ranks and credentials.

However, journal editors may employ schemes that engage junior research fellows as co-reviewers along with their mentors and senior fellows [ 15 ]. Such a scheme is successfully practised within the framework of the Emerging EULAR (European League Against Rheumatism) Network (EMEUNET) where seasoned authors (mentors) train ongoing researchers (mentees) how to evaluate submissions to the top rheumatology journals and select the best evaluators for regular contributors to these journals [ 16 ].

The awareness of the EQUATOR Network reporting standards may help the reviewers to evaluate methodology and suggest related revisions. Statistical skills help the reviewers to detect basic mistakes and suggest additional analyses. For example, scanning data presentation and revealing mistakes in the presentation of means and standard deviations often prompt re-analyses of distributions and replacement of parametric tests with non-parametric ones [ 17 , 18 ].

Constructive reviewer comments

The main goal of the peer review is to support authors in their attempt to publish ethically sound and professionally validated works that may attract readers’ attention and positively influence healthcare research and practice. As such, an optimal reviewer comment has to comprehensively examine all parts of the research and review work ( Table I ). The best reviewers are viewed as contributors who guide authors on how to correct mistakes, discuss study limitations, and highlight its strengths [ 19 ].

Structure of a reviewer comment to be forwarded to authors

Some of the currently practised review models are well positioned to help authors reveal and correct their mistakes at pre- or post-publication stages ( Table II ). The global move toward open science is particularly instrumental for increasing the quality and transparency of reviewer contributions.

Advantages and disadvantages of common manuscript evaluation models

Since there are no universally acceptable criteria for selecting reviewers and structuring their comments, instructions of all peer-reviewed journal should specify priorities, models, and expected review outcomes [ 20 ]. Monitoring and reporting average peer review timelines is also required to encourage timely evaluations and avoid delays. Depending on journal policies and article types, the first round of peer review may last from a few days to a few weeks. The fast-track review (up to 3 days) is practised by some top journals which process clinical trial reports and other priority items.

In exceptional cases, reviewer contributions may result in substantive changes, appreciated by authors in the official acknowledgments. In most cases, however, reviewers should avoid engaging in the authors’ research and writing. They should refrain from instructing the authors on additional tests and data collection as these may delay publication of original submissions with conclusive results.

Established publishers often employ advanced editorial management systems that support reviewers by providing instantaneous access to the review instructions, online structured forms, and some bibliographic databases. Such support enables drafting of evidence-based comments that examine the novelty, ethical soundness, and implications of the reviewed manuscripts [ 21 ].

Encouraging reviewers to submit their recommendations on manuscript acceptance/rejection and related editorial tasks is now a common practice. Skilled reviewers may prompt the editors to reject or transfer manuscripts which fall outside the journal scope, perform additional ethics checks, and minimize chances of publishing erroneous and unethical articles. They may also raise concerns over the editorial strategies in their comments to the editors.

Since reviewer and editor roles are distinct, reviewer recommendations are aimed at helping editors, but not at replacing their decision-making functions. The final decisions rest with handling editors. Handling editors weigh not only reviewer comments, but also priorities related to article types and geographic origins, space limitations in certain periods, and envisaged influence in terms of social media attention and citations. This is why rejections of even flawless manuscripts are likely at early rounds of internal and external evaluations across most peer-reviewed journals.

Reviewers are often requested to comment on language correctness and overall readability of the evaluated manuscripts. Given the wide availability of in-house and external editing services, reviewer comments on language mistakes and typos are categorized as minor. At the same time, non-Anglophone experts’ poor language skills often exclude them from contributing to the peer review in most influential journals [ 22 ]. Comments should be properly edited to convey messages in positive or neutral tones, express ideas of varying degrees of certainty, and present logical order of words, sentences, and paragraphs [ 23 , 24 ]. Consulting linguists on communication culture, passing advanced language courses, and honing commenting skills may increase the overall quality and appeal of the reviewer accomplishments [ 5 , 25 ].

Peer reviewer credits

Various crediting mechanisms have been proposed to motivate reviewers and maintain the integrity of science communication [ 26 ]. Annual reviewer acknowledgments are widely practised for naming manuscript evaluators and appreciating their scholarly contributions. Given the need to weigh reviewer contributions, some journal editors distinguish ‘elite’ reviewers with numerous evaluations and award those with timely and outstanding accomplishments [ 27 ]. Such targeted recognition ensures ethical soundness of the peer review and facilitates promotion of the best candidates for grant funding and academic job appointments [ 28 ].

Also, large publishers and learned societies issue certificates of excellence in reviewing which may include Continuing Professional Development (CPD) points [ 29 ]. Finally, an entirely new crediting mechanism is proposed to award bonus points to active reviewers who may collect, transfer, and use these points to discount gold open-access charges within the publisher consortia [ 30 ].

With the launch of Publons ( http://publons.com/ ) and its integration with Web of Science Group (Clarivate Analytics), reviewer recognition has become a matter of scientific prestige. Reviewers can now freely open their Publons accounts and record their contributions to online journals with Digital Object Identifiers (DOI). Journal editors, in turn, may generate official reviewer acknowledgments and encourage reviewers to forward them to Publons for building up individual reviewer and journal profiles. All published articles maintain e-links to their review records and post-publication promotion on social media, allowing the reviewers to continuously track expert evaluations and comments. A paid-up partnership is also available to journals and publishers for automatically transferring peer-review records to Publons upon mutually acceptable arrangements.

Listing reviewer accomplishments on an individual Publons profile showcases scholarly contributions of the account holder. The reviewer accomplishments placed next to the account holders’ own articles and editorial accomplishments point to the diversity of scholarly contributions. Researchers may establish links between their Publons and ORCID accounts to further benefit from complementary services of both platforms. Publons Academy ( https://publons.com/community/academy/ ) additionally offers an online training course to novice researchers who may improve their reviewing skills under the guidance of experienced mentors and journal editors. Finally, journal editors may conduct searches through the Publons platform to select the best reviewers across academic disciplines.

Peer review ethics

Prior to accepting reviewer invitations, scholars need to weigh a number of factors which may compromise their evaluations. First of all, they are required to accept the reviewer invitations if they are capable of timely submitting their comments. Peer review timelines depend on article type and vary widely across journals. The rules of transparent publishing necessitate recording manuscript submission and acceptance dates in article footnotes to inform readers of the evaluation speed and to help investigators in the event of multiple unethical submissions. Timely reviewer accomplishments often enable fast publication of valuable works with positive implications for healthcare. Unjustifiably long peer review, on the contrary, delays dissemination of influential reports and results in ethical misconduct, such as plagiarism of a manuscript under evaluation [ 31 ].

In the times of proliferation of open-access journals relying on article processing charges, unjustifiably short review may point to the absence of quality evaluation and apparently ‘predatory’ publishing practice [ 32 , 33 ]. Authors when choosing their target journals should take into account the peer review strategy and associated timelines to avoid substandard periodicals.

Reviewer primary interests (unbiased evaluation of manuscripts) may come into conflict with secondary interests (promotion of their own scholarly works), necessitating disclosures by filling in related parts in the online reviewer window or uploading the ICMJE conflict of interest forms. Biomedical reviewers, who are directly or indirectly supported by the pharmaceutical industry, may encounter conflicts while evaluating drug research. Such instances require explicit disclosures of conflicts and/or rejections of reviewer invitations.

Journal editors are obliged to employ mechanisms for disclosing reviewer financial and non-financial conflicts of interest to avoid processing of biased comments [ 34 ]. They should also cautiously process negative comments that oppose dissenting, but still valid, scientific ideas [ 35 ]. Reviewer conflicts that stem from academic activities in a competitive environment may introduce biases, resulting in unfair rejections of manuscripts with opposing concepts, results, and interpretations. The same academic conflicts may lead to coercive reviewer self-citations, forcing authors to incorporate suggested reviewer references or face negative feedback and an unjustified rejection [ 36 ]. Notably, several publisher investigations have demonstrated a global scale of such misconduct, involving some highly cited researchers and top scientific journals [ 37 ].

Fake peer review, an extreme example of conflict of interest, is another form of misconduct that has surfaced in the time of mass proliferation of gold open-access journals and publication of articles without quality checks [ 38 ]. Fake reviews are generated by manipulating authors and commercial editing agencies with full access to their own manuscripts and peer review evaluations in the journal editorial management systems. The sole aim of these reviews is to break the manuscript evaluation process and to pave the way for publication of pseudoscientific articles. Authors of these articles are often supported by funds intended for the growth of science in non-Anglophone countries [ 39 ]. Iranian and Chinese authors are often caught submitting fake reviews, resulting in mass retractions by large publishers [ 38 ]. Several suggestions have been made to overcome this issue, with assigning independent reviewers and requesting their ORCID IDs viewed as the most practical options [ 40 ].

Conclusions

The peer review process is regulated by publishers and editors, enforcing updated global editorial recommendations. Selecting the best reviewers and providing authors with constructive comments may improve the quality of published articles. Reviewers are selected in view of their professional backgrounds and skills in research reporting, statistics, ethics, and language. Quality reviewer comments attract superior submissions and add to the journal’s scientific prestige [ 41 ].

In the era of digitization and open science, various online tools and platforms are available to upgrade the peer review and credit experts for their scholarly contributions. With its links to the ORCID platform and social media channels, Publons now offers the optimal model for crediting and keeping track of the best and most active reviewers. Publons Academy additionally offers online training for novice researchers who may benefit from the experience of their mentoring editors. Overall, reviewer training in how to evaluate journal submissions and avoid related misconduct is an important process, which some indexed journals are experimenting with [ 42 ].

The timelines and rigour of the peer review may change during the current pandemic. However, journal editors should mobilize their resources to avoid publication of unchecked and misleading reports. Additional efforts are required to monitor published contents and encourage readers to post their comments on publishers’ online platforms (blogs) and other social media channels [ 43 , 44 ].

The authors declare no conflict of interest.

Peer Reviewed Literature

What is peer review, terminology, peer review what does that mean, what types of articles are peer-reviewed, what information is not peer-reviewed, what about google scholar.

• How do I find peer-reviewed articles?
• Scholarly vs. Popular Sources

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For more help on this topic, please contact our Research Help Desk: [email protected] or 781-768-7303. Stay up-to-date on our current hours . Note: all hours are EST.

This Guide was created by Carolyn Swidrak (retired).

Research findings are communicated in many ways.  One of the most important ways is through publication in scholarly, peer-reviewed journals.

Research published in scholarly journals is held to a high standard.  It must make a credible and significant contribution to the discipline.  To ensure a very high level of quality, articles that are submitted to scholarly journals undergo a process called peer-review.

Once an article has been submitted for publication, it is reviewed by other independent, academic experts (at least two) in the same field as the authors.  These are the peers.  The peers evaluate the research and decide if it is good enough and important enough to publish.  Usually there is a back-and-forth exchange between the reviewers and the authors, including requests for revisions, before an article is published. 

Peer review is a rigorous process but the intensity varies by journal.  Some journals are very prestigious and receive many submissions for publication.  They publish only the very best, most highly regarded research. 

The terms scholarly, academic, peer-reviewed and refereed are sometimes used interchangeably, although there are slight differences.

Scholarly and academic may refer to peer-reviewed articles, but not all scholarly and academic journals are peer-reviewed (although most are.)  For example, the Harvard Business Review is an academic journal but it is editorially reviewed, not peer-reviewed.

Peer-reviewed and refereed are identical terms.

From  Peer Review in 3 Minutes  [Video], by the North Carolina State University Library, 2014, YouTube (https://youtu.be/rOCQZ7QnoN0).

Peer reviewed articles can include:

• Original research (empirical studies)
• Review articles
• Systematic reviews
• Meta-analyses

There is much excellent, credible information in existence that is NOT peer-reviewed.  Peer-review is simply ONE MEASURE of quality. 

Much of this information is referred to as "gray literature."

Government Agencies

Government websites such as the Centers for Disease Control (CDC) publish high level, trustworthy information.  However, most of it is not peer-reviewed.  (Some of their publications are peer-reviewed, however. The journal Emerging Infectious Diseases, published by the CDC is one example.)

Conference Proceedings

Papers from conference proceedings are not usually peer-reviewed.  They may go on to become published articles in a peer-reviewed journal. 

Dissertations

Dissertations are written by doctoral candidates, and while they are academic they are not peer-reviewed.

Many students like Google Scholar because it is easy to use.  While the results from Google Scholar are generally academic they are not necessarily peer-reviewed.  Typically, you will find:

• Peer reviewed journal articles (although they are not identified as peer-reviewed)
• Unpublished scholarly articles (not peer-reviewed)
• Masters theses, doctoral dissertations and other degree publications (not peer-reviewed)
• Book citations and links to some books (not necessarily peer-reviewed)
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• URL: https://libguides.regiscollege.edu/peer_review

What is peer review?

From a publisher’s perspective, peer review functions as a filter for content, directing better quality articles to better quality journals and so creating journal brands.

Running articles through the process of peer review adds value to them. For this reason publishers need to make sure that peer review is robust.

Editor Feedback

"Pointing out the specifics about flaws in the paper’s structure is paramount. Are methods valid, is data clearly presented, and are conclusions supported by data?” (Editor feedback)

“If an editor can read your comments and understand clearly the basis for your recommendation, then you have written a helpful review.” (Editor feedback)

Peer Review at Its Best

What peer review does best is improve the quality of published papers by motivating authors to submit good quality work – and helping to improve that work through the peer review process. 

In fact, 90% of researchers feel that peer review improves the quality of their published paper (University of Tennessee and CIBER Research Ltd, 2013).

What the Critics Say

The peer review system is not without criticism. Studies show that even after peer review, some articles still contain inaccuracies and demonstrate that most rejected papers will go on to be published somewhere else.

However, these criticisms should be understood within the context of peer review as a human activity. The occasional errors of peer review are not reasons for abandoning the process altogether – the mistakes would be worse without it.

Improving Effectiveness

Some of the ways in which Wiley is seeking to improve the efficiency of the process, include:

• Reducing the amount of repeat reviewing by innovating around transferable peer review
• Providing training and best practice guidance to peer reviewers
• Improving recognition of the contribution made by reviewers

Visit our Peer Review Process and Types of Peer Review pages for additional detailed information on peer review.

Transparency in Peer Review

Wiley is committed to increasing transparency in peer review, increasing accountability for the peer review process and giving recognition to the work of peer reviewers and editors. We are also actively exploring other peer review models to give researchers the options that suit them and their communities.

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As part of the scholarly publishing process, authors' manuscripts often go through peer review before they are published. Watch the video below to learn about the peer review process. As you watch the video, consider these questions:

• What is the purpose of peer review?
• What are the advantages of this process?
• What are the limitations?

Credit: CSU Dominguez Hills ( CC BY-NC 4.0 ) Run Time: 2:58

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Peer-reviewed journals are also called “refereed” or “juried” journals. They are sometimes called "scholarly" or "academic" journals. The peer review process means that a manuscript is reviewed by others in the same field. These individuals (peers) read and review the manuscript, offering their comments and judgment as to its value. The process is intended to enhance the quality of the publications.

Note: You might find different terminology used to refer to peer-reviewed articles. A professor might ask you to find primary studies, primary research, scholarly articles or peer-reviewed articles. These are all generally referred as the same thing, however, if you need further help ask your professor or a librarian!

Below is an example of a peer-reviewed journal for Wildlife Management:

Characteristics of Peer Reviewed Journals

• Journal articles are written by experts in the field. 
• Journal articles are often intended for a person with knowledge in a specific discipline: a medical journal is written for doctors, a legal journal for attorneys, etc.
• The author of a journal article is always listed—usually, along with his or her qualifications or brief information about the author.
• Journal articles include a list of references. This allows you to see what the sources are and to check them if you wish, providing you with other possible resources.
• Scholarly journals are often published by a professional organization or society.
• Often, the word “journal” appears in the title. However, this is not always a good clue: Ladies Home Journal, for instance, is a popular magazine.
• Often, a journal article is preceded by an abstract, or summary of the content.
• Journals do not include advertisements.
• Titles of articles in journals are very revealing of content, not just clever or catchy, as is often the case with popular magazines.
• Scholarly journal articles often report on research; they may include theoretical assumptions, methodology, hypotheses, results, and conclusions. 

Example of a Peer-Reviewed Journal Article

Arizona State University. (n.d.). Anatomy of an Article. Retrieved from https://askabiologist.asu.edu/explore/anatomy-of-an-article

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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.

Meryl Brodsky : Communication and Information Studies

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Janelle Hedstrom : Special Education, Curriculum & Instruction, Ed Leadership & Policy ​

Susan Macicak : Linguistics

Imelda Vetter : Dell Medical School

For help in other subject areas, please see the guide to library specialists by subject .

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.

• October 26, 2022 recording
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Peer Review and Primary Literature: An Introduction: Peer Review: What is it?

• Scholarly Journal vs. Magazine
• Peer Review: What is it?
• Finding Peer-Reviewed Articles
• Primary Journal Literature
• Is it Primary Research? How Do I Know?

What is Peer Review?

What Does "Peer Reviewed" Mean? When a journal is peer-reviewed (also called " Refereed "), it means that all articles submitted for publication have gone through a rigorous evaluation.  To ensure that each article meets the highest standards of scholarship, it is evaluated by the editor(s) of the journal.  This is sometimes called " internal review ." In some cases multiple in-house research editors will evaluate the scholarly strength of the article before even considering it for further peer-review.

The editor(s) then enlists the services of other scholars in the same field as the manuscript's author--in other words, that author's peers. This peer-review process is sometimes called " external review ."  These peer scholars offer their view on the quality of the article and its research.  How appropriate and exacting was the research method?  Were the results presented in the best way possible?  Was the literature review thorough?  Does the article make a significant contribution to the scholarship of that field?  Does it meet the scope of this particular journal? There are many criteria for judgement!

The exact peer-review process will vary between publishers.  Additionally, the whole value of peer review, as it now exists, is often hotly debated. Some believe that the Open Access (OA) movement of publishing research on the web and inviting scrutiny and comment will eventually eliminate the need for publisher driven peer-review.

However, as of now, peer-reviewed journal literature is still considered the highest form of scholarship.  Also, your professors will likely say that they want you to use peer-reviewed articles in your paper--sometimes exclusively.

Links to Additional Pages That Discuss the Peer Review Process

Here are a few additional web links for discussions of specific processes and the current controversies:

• View a PowerPoint presentation on the process at the BMJ (British Medical Journal) Group.
• One major publisher of scholarly journals is Elsevier (the publisher behind ScienceDirect).  Here is a page on their peer review policies.  They link to other related articles, blog entries and the like on the bottom half of that page.
• Even to receive a grant to conduct field research, scientists often must go through a peer review.  Here is an example of that process from the National Institutes of Health.

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Peer-Reviewed Literature

• National Center for Health Statistics (NCHS)
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• Centers for Disease Control and Prevention (CDC)
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3. Health Data Sources

Peer-reviewed journal articles have gone through an evaluation process in which journal editors and other expert scholars critically assess the quality and scientific merit of the article and its research. Articles that pass this process are published in the peer-reviewed literature. Peer-reviewed journals may include the research of scholars who have collected their own data using an experimental study design, survey, or various other study methodologies. They also present the work of researchers who have performed novel analyses of existing data sources, such as the ones described in this section.

Leading Health-Related Journals

• The New England Journal of Medicine
• Health Affairs
• Epidemiologic Reviews
• American Journal of Public Health
• The Milbank Quarterly
• Medical Care

There are several benefits of using peer-reviewed literature, including: the process ensures that the quality of the research and validity of the findings are high, information is available on highly-detailed subject matter and complex analyses, and it is easy to search through millions of articles with online databases.

There are limitations to using peer-reviewed literature, however, including: highly-detailed and complex analyses may be irrelevant for users who are simply searching for descriptive statistics and basic measures of public health; may require a subscription to journals or databases to access articles (which can be costly for individuals, although many universities and other organizations provide access to students and faculty); and may be prone to publication bias due to the fact that studies that report significant statistical results are published in favor of those that yield negative or null results.

Peer-reviewed literature is accessible via academic databases that enable users to execute searches across multiple journals. Here are a few examples:

• PubMed® (including MEDLINE®)
• Cumulative Index to Nursing & Allied Health Literature (CINAHL) Database (EBSCOHost)
• ScienceDirect
• Health Business Full Text (EBSCOHost)
• American Psychological Association PSYCInfo
• Web of Science
• Academic Search Complete (EBSCOHost)

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Clinical Psychology Capstone: Literature Review & Peer Review

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• Last Updated: Apr 24, 2024 1:00 PM
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Protocol for a scoping review study on learning plan use in undergraduate medical education

• Anna Romanova   ORCID: orcid.org/0000-0003-1118-1604 1 ,
• Claire Touchie 1 ,
• Sydney Ruller 2 ,
• Victoria Cole 3 &
• Susan Humphrey-Murto 4  

Systematic Reviews volume  13 , Article number:  131 ( 2024 ) Cite this article

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The current paradigm of competency-based medical education and learner-centredness requires learners to take an active role in their training. However, deliberate and planned continual assessment and performance improvement is hindered by the fragmented nature of many medical training programs. Attempts to bridge this continuity gap between supervision and feedback through learner handover have been controversial. Learning plans are an alternate educational tool that helps trainees identify their learning needs and facilitate longitudinal assessment by providing supervisors with a roadmap of their goals. Informed by self-regulated learning theory, learning plans may be the answer to track trainees’ progress along their learning trajectory. The purpose of this study is to summarise the literature regarding learning plan use specifically in undergraduate medical education and explore the student’s role in all stages of learning plan development and implementation.

Following Arksey and O’Malley’s framework, a scoping review will be conducted to explore the use of learning plans in undergraduate medical education. Literature searches will be conducted using multiple databases by a librarian with expertise in scoping reviews. Through an iterative process, inclusion and exclusion criteria will be developed and a data extraction form refined. Data will be analysed using quantitative and qualitative content analyses.

By summarising the literature on learning plan use in undergraduate medical education, this study aims to better understand how to support self-regulated learning in undergraduate medical education. The results from this project will inform future scholarly work in competency-based medical education at the undergraduate level and have implications for improving feedback and supporting learners at all levels of competence.

Scoping review registration:

Open Science Framework osf.io/wvzbx.

Peer Review reports

Competency-based medical education (CBME) has transformed the approach to medical education to focus on demonstration of acquired competencies rather than time-based completion of rotations [ 1 ]. As a result, undergraduate and graduate medical training programs worldwide have adopted outcomes-based assessments in the form of entrustable professional activities (EPAs) comprised of competencies to be met [ 2 ]. These assessments are completed longitudinally by multiple different evaluators to generate an overall impression of a learner’s competency.

In CBME, trainees will progress along their learning trajectory at individual speeds and some may excel while others struggle to achieve the required knowledge, skills or attitudes. Therefore, deliberate and planned continual assessment and performance improvement is required. However, due to the fragmented nature of many medical training programs where learners rotate through different rotations and work with many supervisors, longitudinal observation is similarly fragmented. This makes it difficult to determine where trainees are on their learning trajectories and can affect the quality of feedback provided to them, which is a known major influencer of academic achievement [ 3 ]. As a result, struggling learners may not be identified until late in their training and the growth of high-performing learners may be stifled [ 4 , 5 , 6 ].

Bridging this continuity gap between supervision and feedback through some form of learner handover or forward feeding has been debated since the 1970s and continues to this day [ 5 , 7 , 8 , 9 , 10 , 11 ]. The goal of learner handover is to improve trainee assessment and feedback by sharing their performance and learning needs between supervisors or across rotations. However, several concerns have been raised about this approach including that it could inappropriately bias subsequent assessments of the learner’s abilities [ 9 , 11 , 12 ]. A different approach to keeping track of trainees’ learning goals and progress along their learning trajectories is required. Learning plans (LPs) informed by self-regulated learning (SRL) theory may be the answer.

SRL has been defined as a cyclical process where learners actively control their thoughts, actions and motivation to achieve their goals [ 13 ]. Several models of SRL exist but all entail that the trainee is responsible for setting, planning, executing, monitoring and reflecting on their learning goals [ 13 ]. According to Zimmerman’s SRL model, this process occurs in three stages: forethought phase before an activity, performance phase during an activity and self-reflection phase after an activity [ 13 ]. Since each trainee leads their own learning process and has an individual trajectory towards competence, this theory relates well to the CBME paradigm which is grounded in learner-centredness [ 1 ]. However, we know that medical students and residents have difficulty identifying their own learning goals and therefore need guidance to effectively partake in SRL [ 14 , 15 , 16 , 17 ]. Motivation has also emerged as a key component of SRL, and numerous studies have explored factors that influence student engagement in learning [ 18 , 19 ]. In addition to meeting their basic psychological needs of autonomy, relatedness and competence, perceived learning relevance through meaningful learning activities has been shown to increase trainee engagement in their learning [ 19 ].

LPs are a well-known tool across many educational fields including CBME that can provide trainees with meaningful learning activities since they help them direct their own learning goals in a guided fashion [ 20 ]. Also known as personal learning plans, learning contracts, personal action plans, personal development plans, and learning goals, LPs are documents that outline the learner’s roadmap to achieve their learning goals. They require the learner to self-identify what they need to learn and why, how they are going to do it, how they will know when they are finished, define the timeframe for goal achievement and assess the impact of their learning [ 20 ]. In so doing, LPs give more autonomy to the learner and facilitate objective and targeted feedback from supervisors. This approach has been described as “most congruent with the assumptions we make about adults as learners” [ 21 ].

LP use has been explored across various clinical settings and at all levels of medical education; however, most of the experience lies in postgraduate medical education [ 22 ]. Medical students are a unique learner population with learning needs that appear to be very well suited for using LPs for two main reasons. First, their education is often divided between classroom and clinical settings. During clinical training, students need to be more independent in setting learning goals to meet desired competencies as their education is no longer outlined for them in a detailed fashion by the medical school curriculum [ 23 ]. SRL in the workplace is also different than in the classroom due to additional complexities of clinical care that can impact students’ ability to self-regulate their learning [ 24 ]. Second, although most medical trainees have difficulty with goal setting, medical students in particular need more guidance compared to residents due to their relative lack of experience upon which they can build within the SRL framework [ 25 ]. LPs can therefore provide much-needed structure to their learning but should be guided by an experienced tutor to be effective [ 15 , 24 ].

LPs fit well within the learner-centred educational framework of CBME by helping trainees identify their learning needs and facilitating longitudinal assessment by providing supervisors with a roadmap of their goals. In so doing, they can address current issues with learner handover and identification as well as remediation of struggling learners. Moreover, they have the potential to help trainees develop lifelong skills with respect to continuing professional development after graduation which is required by many medical licensing bodies.

An initial search of the JBI Database, Cochrane Database, MEDLINE (PubMed) and Google Scholar conducted in July–August 2022 revealed a paucity of research on LP use in undergraduate medical education (UGME). A related systematic review by van Houten–Schat et al. [ 24 ] on SRL in the clinical setting identified three interventions used by medical students and residents in SRL—coaching, LPs and supportive tools. However, only a couple of the included studies looked specifically at medical students’ use of LPs, so this remains an area in need of more exploration. A scoping review would provide an excellent starting point to map the body of literature on this topic.

The objective of this scoping review will therefore be to explore LP use in UGME. In doing so, it will address a gap in knowledge and help determine additional areas for research.

This study will follow Arksey and O’Malley’s [ 26 ] five-step framework for scoping review methodology. It will not include the optional sixth step which entails stakeholder consultation as relevant stakeholders will be intentionally included in the research team (a member of UGME leadership, a medical student and a first-year resident).

Step 1—Identifying the research question

The overarching purpose of this study is to “explore the use of LPs in UGME”. More specifically we seek to achieve the following:

Summarise the literature regarding the use of LPs in UGME (including context, students targeted, frameworks used)

Explore the role of the student in all stages of the LP development and implementation

Determine existing research gaps

Step 2—Identifying relevant studies

An experienced health sciences librarian (VC) will conduct all searches and develop the initial search strategy. The preliminary search strategy is shown in Appendix A (see Additional file 2). Articles will be included if they meet the following criteria [ 27 ]:

Participants

Medical students enrolled at a medical school at the undergraduate level.

Any use of LPs by medical students. LPs are defined as a document, usually presented in a table format, that outlines the learner’s roadmap to achieve their learning goals [ 20 ].

Any stage of UGME in any geographic setting.

Types of evidence sources

We will search existing published and unpublished (grey) literature. This may include research studies, reviews, or expert opinion pieces.

Search strategy

With the assistance of an experienced librarian (VC), a pilot search will be conducted to inform the final search strategy. A search will be conducted in the following electronic databases: MEDLINE, Embase, Education Source, APA PsycInfo and Web of Science. The search terms will be developed in consultation with the research team and librarian. The search strategy will proceed according to the JBI Manual for Evidence Synthesis three-step search strategy for reviews [ 27 ]. First, we will conduct a limited search in two appropriate online databases and analyse text words from the title, abstracts and index terms of relevant papers. Next, we will conduct a second search using all identified key words in all databases. Third, we will review reference lists of all included studies to identify further relevant studies to include in the review. We will also contact the authors of relevant papers for further information if required. This will be an iterative process as the research team becomes more familiar with the literature and will be guided by the librarian. Any modifications to the search strategy as it evolves will be described in the scoping review report. As a measure of rigour, the search strategy will be peer-reviewed by another librarian using the PRESS checklist [ 28 ]. No language or date limits will be applied.

Step 3—Study selection

The screening process will consist of a two-step approach: screening titles/abstracts and, if they meet inclusion criteria, this will be followed by a full-text review. All screening will be done by two members of the research team and any disagreements will be resolved by an independent third member of the team. Based on preliminary inclusion criteria, the whole research team will first pilot the screening process by reviewing a random sample of 25 titles/abstracts. The search strategy, eligibility criteria and study objectives will be refined in an iterative process. We anticipate several meetings as the topic is not well described in the literature. A flowchart of the review process will be generated. Any modifications to the study selection process will be described in the scoping review report. The papers will be excluded if a full text is not available. The search results will be managed using Covidence software.

Step 4—Charting the data

A preliminary data extraction tool is shown in Appendix B (see Additional file 3 ). Data will be extracted into Excel and will include demographic information and specific details about the population, concept, context, study methods and outcomes as they relate to the scoping review objectives. The whole research team will pilot the data extraction tool on ten articles selected for full-text review. Through an iterative process, the final data extraction form will be refined. Subsequently, two members of the team will independently extract data from all articles included for full-text review using this tool. Charting disagreements will be resolved by the principal and senior investigators. Google Translate will be used for any included articles that are not in the English language.

Step 5—Collating, summarising and reporting the results

Quantitative and qualitative analyses will be used to summarise the results. Quantitative analysis will capture descriptive statistics with details about the population, concept, context, study methods and outcomes being examined in this scoping review. Qualitative content analysis will enable interpretation of text data through the systematic classification process of coding and identifying themes and patterns [ 29 ]. Several team meetings will be held to review potential themes to ensure an accurate representation of the data. The PRISMA Extension for Scoping Reviews (PRISMA-ScR) will be used to guide the reporting of review findings [ 30 ]. Data will be presented in tables and/or diagrams as applicable. A descriptive summary will explain the presented results and how they relate to the scoping review objectives.

By summarising the literature on LP use in UGME, this study will contribute to a better understanding of how to support SRL amongst medical students. The results from this project will also inform future scholarly work in CBME at the undergraduate level and have implications for improving feedback as well as supporting learners at all levels of competence. In doing so, this study may have practical applications by informing learning plan incorporation into CBME-based curricula.

We do not anticipate any practical or operational issues at this time. We assembled a team with the necessary expertise and tools to complete this project.

Availability of data and materials

All data generated or analysed during this study will be included in the published scoping review article.

Abbreviations

• Competency-based medical education

Entrustable professional activity

• Learning plan
• Self-regulated learning
• Undergraduate medical education

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Acknowledgements

Not applicable.

This study will be supported through grants from the Department of Medicine at the Ottawa Hospital and the University of Ottawa. The funding bodies had no role in the study design and will not have any role in the collection, analysis and interpretation of data or writing of the manuscript.

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Anna Romanova & Claire Touchie

The Ottawa Hospital Research Institute, Ottawa, Canada

Sydney Ruller

The University of Ottawa, Ottawa, Canada

Victoria Cole

The Ottawa Hospital – Riverside Campus, Ottawa, Canada

Susan Humphrey-Murto

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Contributions

AR designed and drafted the protocol. CT and SH contributed to the refinement of the research question, study methods and editing of the manuscript. VC designed the initial search strategy. All authors reviewed the manuscript for final approval. The review guarantors are CT and SH. The corresponding author is AR.

Authors’ information

AR is a clinician teacher and Assistant Professor with the Division of General Internal Medicine at the University of Ottawa. She is also the Associate Director for the internal medicine clerkship rotation at the General campus of the Ottawa Hospital.

CT is a Professor of Medicine with the Divisions of General Internal Medicine and Infectious Diseases at the University of Ottawa. She is also a member of the UGME Competence Committee at the University of Ottawa and an advisor for the development of a new school of medicine at Toronto Metropolitan University.

SH is an Associate Professor with the Department of Medicine at the University of Ottawa and holds a Tier 2 Research Chair in Medical Education. She is also the Interim Director for the Research Support Unit within the Department of Innovation in Medical Education at the University of Ottawa.

CT and SH have extensive experience with medical education research and have numerous publications in this field.

SR is a Research Assistant with the Division of General Internal Medicine at the Ottawa Hospital Research Institute.

VC is a Health Sciences Research Librarian at the University of Ottawa.

SR and VC have extensive experience in systematic and scoping reviews.

Corresponding author

Correspondence to Anna Romanova .

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The authors declare that they have no competing interests.

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Supplementary Information

Additional file 1. prisma-p 2015 checklist., 13643_2024_2553_moesm2_esm.docx.

Additional file 2: Appendix A. Preliminary search strategy [ 31 ].

Additional file 3: Appendix B. Preliminary data extraction tool.

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Romanova, A., Touchie, C., Ruller, S. et al. Protocol for a scoping review study on learning plan use in undergraduate medical education. Syst Rev 13 , 131 (2024). https://doi.org/10.1186/s13643-024-02553-w

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Systematic Reviews

ISSN: 2046-4053

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Sarcopenia and sarcopenic obesity among older adults in the nordic countries: a scoping review

• Fereshteh Baygi 1   na1 ,
• Sussi Friis Buhl 1   na1 ,
• Trine Thilsing 1 ,
• Jens Søndergaard 1 &
• Jesper Bo Nielsen 1  

BMC Geriatrics volume  24 , Article number:  421 ( 2024 ) Cite this article

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Sarcopenia and sarcopenic obesity (SO) are age-related syndromes that may compromise physical and mental health among older adults. The Nordic countries differ from other regions on prevalence of disease, life-style behavior, and life expectancy, which may impact prevalence of sarcopenia and SO. Therefore, the aim of this study is to review the available evidence and gaps within this field in the Nordic countries.

PubMed, Embase, and Web of science (WOS) were searched up to February 2023. In addition, grey literature and reference lists of included studies were searched. Two independent researcher assessed papers and extracted data.

Thirty-three studies out of 6,363 searched studies were included in this scoping review. Overall prevalence of sarcopenia varied from 0.9 to 58.5%. A wide prevalence range was still present for community-dwelling older adults when definition criteria and setting were considered. The prevalence of SO ranged from 4 to 11%, according to the only study on this field. Based on the included studies, potential risk factors for sarcopenia include malnutrition, low physical activity, specific diseases (e.g., diabetes), inflammation, polypharmacy, and aging, whereas increased levels of physical activity and improved dietary intake may reduce the risk of sarcopenia. The few available interventions for sarcopenia were mainly focused on resistance training with/without nutritional supplements (e.g., protein, vitamin D).

The findings of our study revealed inadequate research on SO but an increasing trend in the number of studies on sarcopenia. However, most of the included studies had descriptive cross-sectional design, small sample size, and applied different diagnostic criteria. Therefore, larger well-designed cohort studies that adhere to uniform recent guidelines are required to capture a full picture of these two age-related medical conditions in Nordic countries, and plan for prevention/treatment accordingly.

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The number of older adults with age-related disorders is expected to increase worldwide [ 1 , 2 ]. Sarcopenia and sarcopenic obesity (SO) are both age-related syndromes that may compromise the physical and mental health of older adults and increase their need for health care services in old age [ 3 , 4 ], and this may challenge the sustainability of health care systems economically and by shortage of health care personnel [ 5 ].

Sarcopenia is characterized by low muscle mass in combination with low muscle strength [ 4 ]. SO is characterized by the co-existence of obesity (excessive adipose tissue) and sarcopenia [ 3 ]. Sarcopenia and SO are both associated with physical disability, risk of falls, morbidity, reduced quality of life and early mortality [ 4 , 6 , 7 , 8 , 9 ]. In SO the consequences of sarcopenia and obesity are combined and maximized [ 4 , 6 , 7 , 8 ].

Etiology of sarcopenia and SO is multifactorial and closely linked to multimorbidity [ 3 , 7 , 8 , 9 , 10 ]. Nevertheless, lifestyle and behavioral components particularly diet and physical activity, are important interrelated factors that potentially can be modified. Physical inactivity and sedentary behavior may accelerate age-related loss of muscle mass, reduce energy expenditure, and increase risk of obesity [ 3 , 11 ]. In addition, weight cycling (the fluctuations in weight following dieting and regain) and an unbalanced diet (particularly inadequate protein intake) may accelerate loss of muscle mass and increase severity of sarcopenia and SO in older adults [ 3 , 12 ]. International guideline for the treatment of sarcopenia emphasizes the importance of resistance training potentially in combination with nutritional supplementation to improve muscle mass and physical function [ 13 ]. Similar therapeutic approach is suggested for treatment of SO [ 14 ]. However, more research is needed to confirm optimal treatment of SO [ 14 ].

According to a recently published meta-analysis the global prevalence of sarcopenia ranged from 10 to 27% in populations of older adults ≥ 60 years [ 15 ]. Further the global prevalence of SO among older adults was 11% [ 8 ]. So, sarcopenia and SO are prevalent conditions, with multiple negative health outcomes and should be given special attention [ 16 ]. Despite the large burden on patients and health care systems, the awareness of the importance of skeletal muscle maintenance in obesity is low among clinicians and scientists [ 3 , 16 ].

A recent meta-analysis on publication trends revealed that despite an increase in global research on sarcopenia, the Nordic countries were only limitedly represented [ 6 ]. Nordic countries may differ from other regions on aspects associated with the prevalence and trajectory of sarcopenia and SO and challenge the representativeness of research findings from other parts of the world. These include a different prevalence pattern of noncommunicable diseases [ 17 ], different life-style behavior and life-style associated risk factors [ 15 , 18 ], and higher life expectancy [ 18 ].

The Nordic countries including Sweden, Finland, Iceland, Norway, Denmark, and three autonomous areas (Åland Islands, Greenland and Faroe Islands) share common elements of social and economic policies such as a comprehensive publicly financed health care system [ 18 , 19 ]. Additionally, these countries have a strong tradition of collaboration including a common vision of a socially sustainable region by promoting equal health and inclusive participation in society for older adults [ 20 ]. Therefore, more insight into the etiology, prevalence, and risk factors for sarcopenia and SO among older adults is a prerequisite for the development and implementation of effective strategies to prevent and treat these complex geriatric conditions in this geographic region. So, the aim of this study is to conduct a scoping review to systematically identify and map the available evidence while also addressing knowledge gaps and exploring the following research questions: (1) What are the prevalence of sarcopenia and SO in older adults living in the Nordic countries? (2) Which risk factors or contributing conditions are involved in the development of sarcopenia and SO in the Nordic Countries? (3) Which interventions to prevent or counteract negative health outcomes of sarcopenia and SO have been tested or implemented among older adults living in the Nordic countries?

Identification of relevant studies

The development and reporting of this review were done by following the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) guidelines [ 21 ].

The literature search was developed to target three main areas: Sarcopenia, sarcopenic obesity, and aging (See Appendix 1 for full search strategy). All studies published before the end of February 2023 were included in this scoping review. The optimal sensitivity of search was obtained by simultaneous search of the following databases: PubMed, Embase, and Web of science (WOS). Additionally, a detailed search for grey literature was performed in relevant databases (e.g., Research Portal Denmark, Libris, Oria, Research.fi). Besides, reference lists of the included studies were reviewed to identify eligible studies. Duplicates and non-peer reviewed evidence (e.g., PhD thesis) were excluded but if the latter contained published articles of relevance, these were included. If more than one publication on similar outcomes (e.g., prevalence) were based on a single study, just one publication was included. Data were extracted from large studies with combined data from several countries only when findings were presented separately for the Nordic countries.

Inclusion and exclusion criteria

The inclusion criteria were as follow : Broad selection criteria were used to be comprehensive: (1) studies with any outcome (e.g., prevalence, risk factors, etc.) to address our research questions on sarcopenia and SO, (2) studies on subjects with age ≥ 60 years in any type of settings (e.g., community, nursing homes, general practice, hospital, outpatients, homecare, etc.), (3) studies using any definition of sarcopenia and SO without restriction for criteria and cutoff values, (4) all type of study designs (e.g., randomized control trials, cohort studies, cross-sectional, etc.), (5) studies should be conducted in the Nordic countries The exclusion criteria are as follow : (1) studies without relevant outcome to sarcopenia or SO, (2) studies without sufficient information to determine eligibility.

Study selection and data extraction

Two independent researchers screened literature and conducted data extraction. Any discrepancies between them were resolved through discussion.

First, duplicates were removed by using EndNote 20.6 software, then titles and abstracts were screened to narrow down the list of potentially eligible studies. Finally, the full text review was done to examine in detail the studies that were not excluded in first step. For more clarification, the reasons for the exclusion were recorded (Fig.  1 ).

PRISMA diagram for searching resources

The following information was extracted: (1) study characteristics (e.g., first author’s name, country, year of publication), (2) characteristics of the target population (e.g., age, sex), (3) study design, setting, intervention duration and follow-up time (if applicable), measurements, tools, criteria, and results.

Study selection

A combined total of 6,358 studies were identified through the initial electronic database and grey literature searches. An additional five articles were identified through other sources (citation searching). After removing duplication, 3,464 articles remained. A total of 3107 articles were excluded based on screening titles and abstracts. Out of the remaining 357 studies, 324 were excluded after the full-text review. Finally, 33 studies met our inclusion criteria and were included in this current scoping review [ 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 ] (Fig.  1 ).

Study characteristics

Table  1 summarized characteristics of the included studies.

The number of documents showed an increasing trend between 2020 and 2021. A peak in the number of publications was observed in 2021 (24.2% of all documents). All the studies were conducted across four (Denmark, Norway, Sweden, and Finland) out of the five Nordic countries and three autonomous areas. The highest contribution in this field was made by Sweden ( n  = 12).

Most studies were conducted in community-dwelling settings [ 22 , 23 , 24 , 28 , 30 , 31 , 35 , 36 , 38 , 39 , 40 , 42 , 45 , 46 , 47 , 48 , 49 , 54 ]. Seven studies included patients with acute diseases (hospital-setting) [ 26 , 27 , 33 , 37 , 50 , 51 , 52 ], while four studies included patients with chronic conditions (out-patient setting) [ 25 , 32 , 41 , 44 ], and one study including nursing-home residents [ 34 ]. In terms of study design, most of the studies were observation studies with a cross-sectional or longitudinal design ( 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 33 , 34 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 ), while three studies [ 32 , 35 , 46 ] applied interventions. It appears, however, that one study [ 32 ] out of the above three interventions is sub-project conducted within the framework of larger intervention program. Sample size ranged from 49 in a cross-sectional case control study [ 52 ] to 3334 in a cohort study [ 30 ].

Five studies were among males only [ 22 , 24 , 36 , 45 , 53 ] and three studies included females only [ 38 , 47 , 54 ]. The rest of the studies had a mixed sample. Top subject area was sarcopenia (31 out of the 33 included studies), and on this subject, publications were categorized into the following research areas (with some studies addressing more areas): prevalence [ 22 , 23 , 24 , 25 , 26 , 27 , 29 , 30 , 33 , 35 , 36 , 37 , 40 , 42 , 44 , 45 , 47 , 49 , 50 , 51 , 52 , 53 , 54 ], risk factors [ 24 , 27 , 28 , 30 , 31 , 34 , 38 , 40 , 42 , 44 , 47 , 49 , 50 , 51 ], and effectiveness of interventions on sarcopenia or indicator of sarcopenia [ 32 , 35 , 46 ].

In most studies sarcopenia was defined according to the criteria set by the European Working Group on Sarcopenia in Older People in the updated version from 2019 (EWGSOP2) ( n  = 15) or the original version from 2010 (EWGSOP) ( n  = 14). However, in some studies multiple criteria such as EWGSOP, EWGSOP2, and National Institutes of Health Sarcopenia Project definition (FNIH) were applied [ 27 , 39 , 43 ], and in other studies alternative criteria were used [ 26 , 33 , 35 , 45 , 57 ].

Different assessment methods of muscle mass including Dual energy X-ray absorptiometry (DXA) [ 22 , 24 , 25 , 27 , 29 , 30 , 32 , 33 , 38 , 39 , 40 , 41 , 45 , 46 , 47 , 52 , 53 , 54 ], Bioelectrical Impedance Analysis (BIA) [ 28 , 31 , 34 , 44 , 48 , 49 ], Bioimpedance Spectroscopy (BIS) [ 35 , 42 , 43 ], Computed Tomography (CT) [ 33 ], and Computed Tomography Angiogram (CTA) [ 26 ] were used in the included studies.

SO were defined by the co-existence of sarcopenia with obesity. Studies on SO used the EWGSOP2 criteria [ 39 ], or the EWGSOP2 criteria for hand grip strength only (probable sarcopenia) [ 23 ] in combination with obesity estimated from BMI cut points [ 23 , 39 ], waist circumference [ 23 , 39 ], and fat mass percentage [ 39 ]. Lastly, one study used measures of body composition measures that reflect adiposity as estimates of SO [ 48 ].

Four studies reported the prevalence of “probable sarcopenia” [ 23 , 30 , 36 , 45 ], while two studies reported the prevalence of sarcopenia and comorbidities (e.g., osteopenia, pre-frailty, malnutrition) [ 33 , 40 ].

Narrative synthesis

Due to the heterogeneity of the studies in definition of sarcopenia, settings, and sample size, the overall reported prevalence was variable and ranged from 0.9% [ 54 ] to 58.5% [ 26 ]. However, according to the most commonly used criteria (EWGSOP2) the highest (46%) and lowest (1%) prevalence of sarcopenia was reported in Sweden among inpatients in geriatric care [ 27 ], and community-dwelling older adults [ 30 ], respectively.

Prevalence of sarcopenia according to population and definition criteria is illustrated in Table  2 . Higher prevalence rates of sarcopenia were found in females compared to males among community-dwelling older adults [ 49 ] and in older adults acutely admitted to hospital [ 51 ]. Further, acutely admitted female patients also presented with more severe sarcopenia compared to male patients [ 51 ].

Frequency of sarcopenia was higher (9.1–40.0%) in patients with diabetes (with and without complications of charcot osteoarthropathy), compared to age-matched healthy adults [ 52 ].

The prevalence of “probable sarcopenia” ranged between 20.4% (reduced muscle strength only) and 38.1% (fulfilling one of the following criteria: reduced muscle strength, reduced muscle mass, or low physical function) in Finnish community-dwelling adults [ 23 , 36 ], while longitudinal studies on Swedish community-dwelling old (70 years) and very old adults (≥ 85 years) the prevalence of “probable sarcopenia” (reduced muscle strength only) ranged from 1.8 to 73%, respectively [ 30 , 45 ]. Lastly, in a Swedish study among nursing home residents the prevalence of probable sarcopenia was 44% (evaluated by an impaired chair stand test) [ 34 ].

Prevalence of Osteosarcopenia (sarcopenia and osteoporosis) was 1.5% [ 36 ], and the prevalence of co-occurrence of all three following conditions: pre-frail, malnutrition, and sarcopenia was 7% [ 34 ].

We only identified two studies with prevalence of SO [ 39 ] and probable SO [ 23 ]. The prevalence of SO in a Swedish population was 4% and 11% in females and males, respectively, while the prevalence of probable SO among Finnish community-dwelling ranged between 5.8% and 12.6%, depending on the criteria to define the obesity (e.g., BMI, waist circumference, etc.) [ 23 ].

Several studies investigated aspects of etiology and risk factors for sarcopenia [ 24 , 27 , 28 , 30 , 31 , 34 , 36 , 38 , 40 , 42 , 43 , 44 , 47 , 49 , 50 , 51 ] and one study focused on SO [ 49 ]. Higher physical activity was associated with a decreased likelihood of sarcopenia [ 30 ]. In addition, adhering to world health organization (WHO) guidlines for physical activity and the Nordic nutritional recommendations for protein intake was positively associated with greater physical function and lower fat mass in older female community-dwellers [ 38 ]. In older adults who are physically active, eating a healthy diet (based on the frequency of intake of favorable food like fish, fruits, vegetables, and whole grains versus unfavorable foods like red/processed meats, desserts/sweets/sugar-sweetened beverages, and fried potatoes) was associated with lower risk of sarcopenia [ 28 ]. Further, among older adults who already meet the physical activity guidelines, additional engagement in muscle-strengthening activities was associated with a lower sarcopenia risk score and improved muscle mass and chair rise time [ 31 ].

Associations between sarcopenia, risk of sarcopenia and malnutrition or nutritional status was identified in geriatric patients [ 27 , 51 ], older patients with hip fracture [ 50 ], nursing home residents [ 34 ] and in community-dwelling older adults [ 49 ]. Moreover, the importance of nutritional intake was investigated in the following studies [ 24 , 36 , 47 ]. A study among community-dwelling men revealed an inverse association between total energy intake, protein intake (total, plant, and fish protein), intake of dietary fibers, fat (total and unsaturated), and vitamin D with sarcopenia status [ 36 ]. In a cohort of 71-year-old men a dietary pattern characterized by high consumption of fruit, vegetables, poultry, rice and pasta was associated with lower prevalence of sarcopenia after 16 years [ 24 ]. A longitudinal Finnish study on sarcopenia indices among postmenopausal older women, showed that lower adherence to the Mediterranean (focuses on high consumption of olive oil) or Baltic Sea (focuses on the dietary fat quality and low-fat milk intake) diets resulted in higher loss of lean mass over a 3-year period [ 47 ]. Further, a higher adherence to the Baltic Sea diet was associated with greater lean mass and better physical function, and higher adherence to the Mediterranean diet was associated with greater muscle quality [ 47 ].

In a study of patients with hip fracture age, polypharmacy, and low albumin levels was associated with sarcopenia [ 50 ]. Exocrine pancreatic insufficiency was an independent risk factor for sarcopenia [ 44 ]. This study also revealed that sarcopenia was associated with reduced quality of life, physical function, and increased risk of hospitalization [ 44 ]. In a longitudinal study of community-dwelling adults (+ 75 years) at risk of sarcopenia, high physical function, muscle strength, muscle mass and low BMI predicted better physical function and reduced need for care after four years [ 42 ]. Furthermore, in community-dwelling adults with sarcopenia, muscle mass, muscle strength and physical function are independent predictors of all-cause mortality. As a result, they have been proposed by researchers as targets for the prevention of sarcopenia-related over-mortality [ 43 ]. Lastly, community-dwelling older adults with sarcopenia had lower bone mineral density compared to those without sarcopenia and they were more likely to develop osteoporosis (Osteosarcopenia) [ 40 ].

Regarding SO risk factors, a longitudinal study among community-dwelling older adults in Finland found that SO (operationalized by measures of adiposity) were associated with poorer physical function after ten years [ 48 ].

Our literature search identified three randomized controlled trials investigating the effectiveness of interventions to prevent or counteract sarcopenia in older adults of Norway, Finland, and Sweden, respectively [ 32 , 35 , 46 ]. The Norwegian study [ 32 ] was a double-blinded randomized controlled trial (RCT). The study included those who were at risk of developing sarcopenia, including patients with chronic obstructive pulmonary disease (COPD) or individuals who showed diagnostic indications of sarcopenia. Participants received either vitamin D 3 or placebo supplementation for 28 weeks. Additionally, resistance training sessions were provided to all participants from weeks 14 to 27. Vitamin D supplementation did not significantly affect response to resistance training in older adults at risk of sarcopenia with or without COPD [ 32 ].

Furthermore, a RCT among pre-sarcopenic Swedish older adults investigated the effectiveness of three weekly sessions of instructor-led progressive resistance training in combination with a non-mandatory daily nutritional supplement (175 kcal, 19 g protein) compared to control group. The 10 weeks intervention resulted in significant between group improvements of physical function and a significant improvement in body composition in the intervention group [ 46 ].

Another intervention study revealed that a 12-month intervention with two daily nutritional supplements (each containing 20 g whey protein) did not attenuate the deterioration of physical function and muscle mass in sarcopenic older community-dwelling adults compared to isocaloric placebo supplements or no supplementation. All participants were given instructions on home-based exercises, importance of dietary protein and vitamin D supplementation [ 35 ].

Based on our broad literature search 33 studies were identified that concerned sarcopenia and SO and met the inclusion criteria. However, research on SO was very limited with only three studies identified. Narrative synthesis of the included studies revealed that the most reported classification tool for sarcopenia in Nordic countries was the EWGSOP2. Moreover, some studies estimated sarcopenia using EWGSOP. The overall prevalence of sarcopenia in Nordic countries according to EWGSOP2 ranged between 1% and 46% [ 25 , 28 ]. The prevalence of SO, however, was reported only in one study in Sweden (4–11%) [ 39 ]. Even though the previous systematic reviews and meta-analysis have reported the prevalence of sarcopenia and SO in different regions and settings (e.g., community-dwelling, nursing home, etc.) [ 8 , 15 , 55 , 56 ], this current scoping review is to the best of our knowledge the first study that provides an overview of research on sarcopenia and SO in the Nordic countries.

Based on our findings from 24 studies, there were large variability in prevalence of sarcopenia in studies conducted in the Nordic countries. We think that the wide variation in estimated prevalence of sarcopenia in our scoping review might be due to a different definition/diagnostic criterion (e.g., EWGSOP, EWGSOP2, FNIH), methodology to measure muscle mass (DXA, BIA, CT), and heterogeneity in characteristics of the study population (e.g., setting, age, medical conditions, co-occurrence of multiple risk factors). A previous study on prevalence of sarcopenia in Swedish older people showed significant differences between prevalence of sarcopenia based on EWGSOP2 and EWGSOP1 [ 29 ]. Therefore, researchers stressed that prevalence is more dependent on cut-offs than on the operational definition [ 29 , 57 ]. Further, we know that various international sarcopenia working groups have issued expert consensus and such diagnostic criteria are being updated [ 4 , 58 ]. Since the revision of criteria focuses primarily on the adjustment of cut-off values, the main reason for differences in prevalence even when using an updated version of one diagnosis criteria is modification in cut-off values. For instance, if the cut-off value for gait speed was increased by 0.2 m/s, the prevalence of sarcopenia may increase by 8.5% [ 57 ]. Meaning that even a small change in cut-off value can have a big impact on how sarcopenia is diagnosed. Besides when we take definition criteria into account (Table  2 ), the prevalence of sarcopenia is still variable in the population of community-dwelling adults for instance. We believe it is basically because studies have applied different assessment tools and tests to identify older adults with low muscle mass and muscle strength, although using the same definition criteria (Table  1 ). Previous studies have illustrated that choice of methodology to assess muscle strength (e.g., hand grip strength, chair rise) [ 59 ] and muscle mass (e.g., DXA, BIA, anthropometry) [ 60 , 61 , 62 ] in older adults may impact findings and this variability may explain some of the variability in our findings. So, adherence to the latest uniform diagnostic criteria for future studies is recommended to simplify the comparison of findings within the same country, across countries, and regions. Moreover, we suggest that medical community particularly GPs to come to an agreement on assessment methods for muscle mass and muscle strength and the use of one set of definition criteria for sarcopenia.

In previous meta-analyses [ 15 ], sub-group analyses based on region and classification tool, revealed that the prevalence of sarcopenia was higher in European studies using EWGSOP (12%) compared to rest of the studies using Asian Working Group for Sarcopenia (AWGS), FNIH, and EWGSOP (3%) [ 15 ]. In our scoping review, we also found a high prevalence of sarcopenia in Nordic countries. Longevity and life expectancy is higher in the Nordic countries compared to estimates for rest of the world [ 18 ], which means that in this region many people reach old age, and consequently they are more likely to be diagnosed with sarcopenia as an age-related disorder. Therefore, the authors of this current scoping review emphasis the importance of preventive strategies targeted major risk factors and effective interventions to limit the consequences of sarcopenia in the Nordic populations. Besides, we think that the health care system in the Nordic countries should be better equipped with the necessary healthcare resources for both a timely diagnosis and dealing with this major age-related issue in the years to come. However, due to the limitations regarding the timely diagnosis, we highly recommend a comprehensive approach including establishment of support services, implement educational programs, offer training for health care professionals, and engage the community.

Many countries have conducted research on SO [ 7 , 39 , 63 , 64 , 65 ]. Based on our findings, however, among the Nordic countries only Sweden and Finland have investigated the prevalence of probable SO and SO [ 23 , 29 ]. Besides, we only found one study investigating the association between body adiposity and physical function over time [ 54 ]. We did not find any literature on risk factors or interventions among older adults with SO in this region. Therefore, we call on medical and research community in Nordic countries to attach importance to screening of SO in elderly people to capture a full picture of this public health risk to aging society and allocate healthcare resources accordingly.

In terms of risk factors for sarcopenia, our study revealed that malnutrition, low levels of physical activity, specific diseases (e.g., diabetes, osteoporosis), inflammation, polypharmacy (multiple medicines), BMI, and ageing are potential risk factor for sarcopenia in populations of the Nordic region. However, evidence on risk factors derived mainly from cross-sectional associations [ 27 , 28 , 30 , 31 , 34 , 40 , 44 , 49 , 50 , 51 ], and only to a limited extend from longitudinal studies [ 24 , 38 , 43 , 47 ]. Therefore, the associations between risk factors and sarcopenia should be interpreted with caution due to the possibility of reverse causality and confounding affecting the results. Moreover, our findings on risk factors mainly came from community-dwelling older adults, and only to a limited extend hospital and nursing home settings. We think that risk factors may vary depending on population characteristics (e.g., age, sex, health condition) and setting (e.g., hospital, nursing home, community). Therefore, we encourage researchers of the Nordic countries to perform well-designed prospective cohort studies in different settings to enhance the possibility to establish causal inference as well as understanding degree and direction of changes over time.

A recently published meta-analyses revealed a higher risk of having polypharmacy in Europe among individuals with sarcopenia compared to people without this condition [ 66 ]. A nationwide register-based study in Swedish population also showed that the prevalence of polypharmacy has increased in Sweden over the last decade [ 67 ]. Sarcopenia itself is associated with morbidity (identified by specific disease or inflammatory markers) and different health-related outcomes (e.g., disability) [ 7 ]; therefore, future research should investigate whether polypharmacy is a major factor to sarcopenia development [ 66 ]. Although we lack information on polypharmacy in Nordic countries other than Sweden, we encourage researchers in this region to examine the above research gap in their future studies.

According to previous studies physiological changes in ageing include systemic low-grade inflammation which results in insulin resistance, affect protein metabolism and leads to increased muscle wasting [ 68 ]. Acute and chronic disease may increase the inflammatory response and accelerate age-related loss of muscle mass and increase risk of sarcopenia [ 68 , 69 ]. Hence, we think that special attention should be made by health care professionals particularly GPs to older adults with acute or chronic conditions to limit the risk of sarcopenia.

Literature from the Nordic countries also indicated that higher levels of physical activity and different dietary patterns (e.g., higher protein intake, fruit, vegetables, fibers) were associated with reduced risk of sarcopenia or improvement in indicators of sarcopenia. There was a large heterogeneity in the studied aspect which makes direct comparison of studies difficult. Nevertheless, according to findings from a recent systematic review of meta-analyses on sarcopenia the identified risk factors are in alignment with previously identified risk factors globally [ 70 ]. Other potential lifestyle-related risk factors suggested from the above meta-analysis included smoking and extreme sleep duration. However, we did not identify studies investigating these health behaviors in the Nordic populations. Therefore, high-quality cohort studies are needed to deeply understand such associations with the risk of sarcopenia.

In this current review, we only found three intervention studies in Nordic countries. However, two of them were sub-projects of big intervention programs, meaning that such studies were not designed explicitly for the prevention/treatment of sarcopenia. Therefore, explicit intervention studies on sarcopenia in this region is recommended.

We believe that on a global level, research on sarcopenia will carry on with nutrition, exercise, and understanding of molecular mechanisms. Furthermore, examining the link between sarcopenia and other medical conditions/diseases would be the next step [ 6 ]. In the Nordic countries, however, already performed studies have a basic and descriptive design, so that, well-designed research and advanced analyses are lacking. Hence, we recommend conducting large well-designed and adequately powered studies to (a) explore the scale of this age-related health issue on country and regional level, (b) investigate the patterns of physical activity and sedentary behavior to understand if this should be a target in older adults with SO and sarcopenia, (c) determine whether elderly populations are suffering from nutritional deficiency or are at risk of malnutrition. The latest can support further studies to assess the impact of combined physical activity and dietary intake, which are still lacking globally [ 6 ].

A previous systematic review on therapeutic strategies for SO revealed that exercise-based interventions (e.g., resistance training) reduced total adiposity and consequently improved body composition. However, evidence of other therapeutic strategies (e.g., nutritional supplementation) was limited due to scarcity of data and lack of unique definition for SO [ 69 ]. Therefore, authors suggested that more research should be done to clarify optimal treatment options for various age-groups and not only for older adults [ 14 ].

In our scoping review, the included studies, did not provide a status of either SO or the prevention/treatment methods in this region. We believe that SO is practically neglected in clinical practice and research as well, and this is mainly because it is difficult to separate it from general obesity. The consequence of lacking knowledge in this research area is that when older adults with SO are recommended weight loss- a frequently used strategy for management of general obesity- this may accelerate the loss of muscle mass and increase the severity of the sarcopenia [ 3 ]. Consequently, we think that this issue may have adverse effects both on patients (e.g., decreasing quality of their life) and on the health care system (e.g., increasing the health care demands) of this region. Therefore, we encourage researchers to perform cohort studies to understand the epidemiology and etiological basis of SO, which are poorly understood even on a global scale [ 8 ]. We think that the consensus definition on SO from the European Society for Clinical Nutrition and Metabolism (ESPEN) and European Association for the Study of Obesity (EASO) which was published in 2022 [ 3 ], can positively affect the ability to define studies on prevalence and prevention of SO. Besides, we recommend conducting further research to find the optimal treatment for SO and reduce its adverse consequences both at individual and society levels. Additionally, we think that the concepts of sarcopenia and SO might be somehow unfamiliar to health care personnel. Therefore, it is highly recommended that more information be provided to bring their attention to the significance of prevention, timely diagnosis, and treatment of these two aging disorders.

Strengths and limitations of the study

This is the first study providing an overview of available evidence on sarcopenia and SO among older adults in the Nordic countries. These countries have important similarities in welfare sectors and on a population level and we believe that our findings will be a significant benefit for researchers and health care providers to understand the knowledge gaps and plan for future studies in this geographical region. However, the current scoping review has limitations. This review was limited to studies among individuals more than 60 years old which may limit the overview of available research in this field, as well as understanding risk factors, confounders for prevention, and the potential for early detection of these two diseases in younger age population. The included cross-sectional studies in our review cannot provide information on causality of the associations.

Sarcopenia and SO are generally prevalent syndromes among older adults in Nordic countries, even though the prevalence of them varies according to the criteria for definition, population, and setting. Research among older adults with SO was very limited in this region. Besides, studies on risk factors were primarily cross-sectional and only few intervention studies were identified. Therefore, we encourage researchers performing well-designed studies (e.g., prospective cohorts) to understand the epidemiology and etiological basis of these two age-related disorders. For the next step, implementation of interventions targeting risk factors (e.g., combined physical activity and dietary intake) and evaluating of their impact on prevention or treatment of sarcopenia and SO is recommended. Furthermore, for the comprehensive advancement of muscle health in older adults, we recommend implementing interventions directed at health care personnel and encouraging more collaboration among clinicians, professional societies, researchers, and policy makers to ensure comprehensive and effective approach to health care initiatives.

Data availability

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

Abbreviations

sarcopenic obesity

Web of science

Preferred Reporting Items for Systematic Reviews and Meta-analyses

European Working Group on Sarcopenia in Older People in the updated version from 2019

National Institutes of Health Sarcopenia Project definition

Dual energy X-ray absorptiometry

Bioelectrical Impedance Analysis

Bioimpedance Spectroscopy

Computed Tomography

Computed Tomography Angiogram

World Health Organization

General Practitioner

Randomized Controlled Trial

Chronic Obstructive Pulmonary Disease

European Association for the Study of Obesity

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FB conceived and designed the review, participated in literature review, data extraction, interpretation of the results and wrote the manuscript. SFB designed the review, participated in literature review, data extraction, and revised the manuscript. TT, JBN and JS contributed to the conception of the study and revised the manuscript critically. All the authors approved the final manuscript.

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Baygi, F., Buhl, S.F., Thilsing, T. et al. Sarcopenia and sarcopenic obesity among older adults in the nordic countries: a scoping review. BMC Geriatr 24 , 421 (2024). https://doi.org/10.1186/s12877-024-04970-x

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Intestinal perforation due to colorectal cancer during pregnancy: case report and literature review

• Yan Gao 1 &

BMC Pregnancy and Childbirth volume  24 , Article number:  374 ( 2024 ) Cite this article

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Colorectal cancer (CRC) in pregnancy is sporadic. We reported a case of a woman at 23 + 4 weeks of gestation who presented with abdominal pain. The patient underwent an ultrasound and MRI, during which a colonic mass was noted. Considering a probable incomplete intestinal obstruction, a colonoscopy, biopsy, and colonic stenting were performed by a multidisciplinary team. However, sudden hyperthermia and CT demonstrated intestinal perforation, and an emergency caesarean section and colostomy were conducted. The histological analysis confirmed moderately high-grade adenocarcinoma.

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Case presentation

A 36-year-old Chinese woman (gravida 0 para 0) with no family history of cancer presented at the Emergency Department of Peking University First Hospital, Beijing, China, at 23  +4  weeks of pregnancy. She complained of 3-day diarrhoea and aggregating abdominal pain. The patient had iron deficiency anaemia for five years and rectal bleeding for two years, which was treated with sigmoidoscopy and haemorrhoid surgery, and she denied other diseases in her remote pathological history.

Upon admission, physical examination revealed abdominal tenderness and rebound pain, particularly in the right upper quadrant (RUQ) and below the xiphoid. There was a suspicious tenderness at McBurney's point. The laboratory examination showed as follows: white blood cell (WBC) count 10.51X10 9 /L, haemoglobin (Hb) 82 g/L, C-reactive protein (CRP) 40 mg/L, potassium 3.08 mmol/L, sodium 131.21mmo1/L; otherwise, the coagulation function, amylase, lipase, and liver and kidney function were within normal range. Abdominal ultrasound demonstrated dilated intestines at the left upper quadrant and a mass measuring 8 cm behind the uterus suggestive of intestinal origin. The abdominal and pelvic Magnetic Resonance Imaging (MRI) without contrast indicated an irregular thickening of the large bowel between the colon and sigmoid colon, with an extension of 8-10 cm (Fig.  1 ). Therefore, a rectum and sigmoid colon neoplasm accompanied by incomplete intestinal obstruction was the initial suspicion.

MRI at admission

Considering the complexity of the patient's condition, a multidisciplinary meeting was organised, including the department of obstetrics, general surgery, haematology, endoscopy, imaging department, and other departments. During the discussion, all the participating departments agreed with the initial suspicion of colon cancer (T4aN + Mx). A colonoscopy was recommended to clarify the diagnosis further and to decide whether termination of pregnancy was appropriate based on the pathological results.

An emergency sedation-free colonoscopy was performed and revealed an exophytic circumferential mass located at the junction of the rectum and sigmoid colon, 13 cm away from the anus. The mass surface was ulcerated and covered with white moss. Additionally, there was a narrowing lumen of 3-4 cm in length, which did not allow for the endoscope to progress (Fig.  2 ). The biopsy was taken. Meantime, anti-infection therapy, iron intravenous iron supplementation therapy, and parenteral nutrition were initiated.

Colonoscopy findings

On the second day after admission, the patient's pain was relieved. The stool was soft and yellow, with a small amount of red mucus visible on the surface. Since the haemoglobin level gradually decreased to 69 g/L, 2 Units of red blood cells were transfused to correct anaemia. After a blood transfusion, the patient developed a fever, with a maximum temperature of 38.9 ℃, accompanied by chills, shortness of breath, and wheezing (50–60 breaths/minute). Blood oxygen saturation was 91% and 94% after oxygen uptake of 3L/min, blood pressure was 102/60 mmHg. Full blood count (FBC) showed WBC 10.07 × 10 9 /L, Hb 81 g/L, platelet 204 × 10 9 /L, and CRP 146 mg/L. Blood gas analysis showed a pH of 7.46, an oxygen saturation index of 94%, and oxygen partial pressure of 65 mmHg. An emergent computed tomography (CT) suggested intestinal perforation due to colon lesions.

Consequently, at 23 +5  weeks of gestation, an emergency cesarean section and transverse colostomy were performed simultaneously with the collaboration of obstetricians and a colorectal surgeon. A transverse incision was made, and a large lesion occupying the sigmoid colon was detected during the operation, surrounded by enlarged lymph nodes. No metastatic nodules were found in the peritoneum, omentum, and pelvic wall, and no apparent signs of perforation were observed in the intestinal canal above the retroflexion of the peritoneum. A latex drainage tube behind the uterus was placed. After the surgery, the patient was transferred to the Surgical Intensive Care Unit (SICU), where she received treatment including acid suppression, anti-infection therapy and parenteral nutritional support, which gradually transitioned to a regular diet. Eventually, the patient was discharged on the 11th day post-operation.

The postoperative histopathological result demonstrated moderately to high differentiated adenocarcinoma. Immunohistochemical staining results were listed: P53 90% + , Her-20, MLH1 + , PMS2 + , MSH2 + , MSH6 + . Microsatellite stability: MSS. No tumour cells were found in the placenta. Under the guidance of general surgery, the patient took capecitabine orally 25 days after surgery and planned to undergo two courses of intravenous chemotherapy before undergoing further surgical treatment.

Colorectal cancer (CRC) in pregnancy is exceedingly rare. The incidence rate of CRC in pregnancy is 0.002% to 0.008% [ 1 ]. Typical symptoms/signs include hematochezia or melena, abdominal pain, apart from unexplained iron deficiency anaemia, or a change in bowel habits [ 2 ]. Less common presenting symptoms include abdominal distention or nausea and vomiting, which may be indicators of obstruction.

A delayed diagnosis during pregnancy may be due to overlapping symptoms with that of normal pregnancy in the context of the expected low incidence of CRC at such an early maternal age. The reluctance of medical teams to conduct diagnostic tests due to potential risks to the fetus often leads to a delayed diagnosis, thus complicating treatment and worsening the prognosis [ 3 ]. Various serum markers have been associated with CRC, particularly carcinoembryonic antigen (CEA). However, all these markers, including CEA, have a low diagnostic ability to detect primary CRC due to significant overlap with benign disease and low sensitivity for early-stage disease [ 4 ].

Nonionising radiation imaging, such as Ultrasound and MRI, is favoured during pregnancy. Despite low intrauterine doses, CT scan can be used in pregnancy if necessary.

Diagnostic imaging studies typically expose the fetus to less than 50 mGy (0.05 Gy, five rads), and there is no evidence of an increased risk of fetal anomalies, intellectual disability, growth restriction, or pregnancy loss from ionising radiation at this dose level [ 5 , 6 ].

In the United States and elsewhere, the standard practice at most institutions is that all patients with stage II, III, or IV CRC undergo chest, abdomen, and pelvic CT before or after resection, an approach endorsed by the National Comprehensive Cancer Network.

Colonoscopy is the most accurate diagnostic test for CRC since it can localise and biopsy lesions throughout the large bowel, detect synchronous neoplasms and remove polyps. Endoscopy is recommended in pregnancy when the patient has significant or continuous bleeding, severe or refractory nausea and vomiting or abdominal pain, and strong suspicion of colon mass [ 7 ].

The optimal time for advanced endoscopic procedures during the pregnancy is the second trimester; however, if the consequences of a delayed procedure can cause harm to the patient or the fetus, then one should proceed with a multidisciplinary team [ 8 ].

GI endoscopy in pregnant patients is inherently risky because the fetus is susceptible to maternal hypoxia and hypotension, either of which could lead to fetal demise. Other risks to the fetus include teratogenesis (from medications given to the mother or ionising radiation exposure) and premature birth [ 9 ].

A systematic review was conducted to identify studies regarding CRC-p and conduct a pooled analysis of the reported data. Seventy-nine papers written on 119 patients with unequivocal CRC-p were included. The calculated pooled risk is 0.002%, and age at diagnosis has decreased over time. The median age at diagnosis was 32 (range, 17–46) years. 12%, 41% and 47% of CRC-p were diagnosed during the first, second and third trimester respectively. Among the cases, bleeding occurred in 47% of patients, abdominal pain in 37.6%, constipation in 14.1%, obstruction in 9.4% and perforation in 2.4%. Regarding cancer, 53.4% of the CRC-p was in the colon, while 44% was in the rectum. Out of 82 patients whose treatment was described, 9.8% received chemotherapy during pregnancy. None of their newborns developed permanent disability, one developed hypothyroidism, and 72% of newborns were alive. Hence, treatment of CRC-p should be timely and needs to be discussed carefully by a multidisciplinary team, with close patient monitoring [ 10 ].

Treatment of CRC-p is influenced by several factors, including tumor location and stage at presentation, surgical settings (elective vs. urgent/ emergent) and gestational age. The decision-making process must involve anesthesiologists, colorectal surgeons, oncologists and gynaecologists, while the mother will make the final decision.

If the tumour is resectable, surgical excision after diagnosis should be performed as soon as possible if the diagnosis is made before 20 weeks of gestation. A total abdominal hysterectomy may be necessary to provide greater access to the rectum or if the uterus is involved. If diagnosis is made later in pregnancy (> 20 weeks), surgery can be postponed until fetal pulmonary maturity is reached (28–32 weeks) or after delivery. However, waiting until after the fetus is delivered does pose risks to the mother, and the patient should be fully informed of these risks [ 11 ].

When malignant neoplasm is diagnosed during gestation, maternal life-saving chemotherapy poses life-threatening concerns for the developing fetus [ 12 ]. Exposure to chemotherapy in the first trimester poses the most significant risk for teratogenicity, with an incidence of spontaneous abortions or malformations up to 15–25% [ 13 ]. In the second or third trimesters, chemotherapy is generally considered safer but is associated with an increased incidence of small for gestational age fetuses (SGA) [ 14 , 15 ]. Chemotherapy is often continued until 35 gestational weeks or three weeks before the expected due date. Timing is recommended to avoid the increased risk of chemotherapy-related complications, such as bone marrow suppression, bleeding, and maternal and fetal death during delivery [ 16 ].

A distance of > 30 cm from the field edges will expose the embryo/fetus to only 4–20 cGy. Therefore, many areas (e.g., head and neck, breast, and extremities) can be treated with radiation. Lead shielding over the embryo or fetus can also reduce the exposure. Because of the location of the tumour and proximity of the embryo/fetus, Radiation therapy (RT) is contraindicated during pregnancy. Radiation therapy can be used postoperatively only after delivery or elective abortion in pregnant patients. Future fertility should be considered before treatment because RT can cause permanent damage to the ovaries, which can lead to infertility [ 11 , 17 ].

Colorectal cancer is the most common cause of large bowel obstruction, comprising 60 per cent of all cases.

Patients who present with an acute malignant colorectal obstruction may require immediate surgery if they have a perforation or pending perforation, or if they are clinically unstable (tachycardic, hypotensive, acidotic), or are symptomatic. Perforation occurs more commonly at the point of obstruction, most likely due to local tumour invasion or inflammatory reaction, rather than in the proximal, dilated colon. The decision to choose a staged versus one-stage procedure depends upon several factors, including the location of the obstructing lesion, condition of the proximal colon, medical comorbidities of the patient, as well as their life expectancy, goals of care, and the presence of proximal perforation [ 18 , 19 , 20 ].

Colorectal cancer in pregnancy is rare. For patients whose symptoms include chronic anaemia that is difficult to correct with iron supplements and persistent gastrointestinal symptoms during pregnancy, it is necessary to be vigilant about the possibility of digestive tract tumours. It's essential to note that tumours should be suspected when all benign causes are excluded despite low incidence [ 21 , 22 ]. Ultrasound and magnetic resonance imaging are relatively safe examinations during pregnancy, and if necessary, pelvic CT and gastroscopy can be performed for diagnosis.

Availability of data and materials

All data generated or analysed during this study are included in this published article.

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Gao, Y., Sun, Y. Intestinal perforation due to colorectal cancer during pregnancy: case report and literature review. BMC Pregnancy Childbirth 24 , 374 (2024). https://doi.org/10.1186/s12884-024-06533-9

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Severe fever with thrombocytopenia syndrome with central nervous system symptom onset: a case report and literature review

• Dawei Shan 1 ,
• Weibi Chen 1 ,
• Gang Liu 1 ,
• Huimin Zhang 1 ,
• Shuting Chai 1 &
• Yan Zhang 1  

BMC Neurology volume  24 , Article number:  158 ( 2024 ) Cite this article

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Severe fever with thrombocytopenia syndrome (SFTS) is a natural focal disease transmitted mainly by tick bites, and the causative agent is SFTS virus (SFTSV). SFTS can rapidly progress to severe disease, with multiple-organ failure (MOF) manifestations such as shock, respiratory failure, disseminated intravascular coagulation (DIC) and death, but cases of SFTS patients with central nervous system (CNS) symptoms onset and marked persistent involuntary shaking of the perioral area and limbs have rarely been reported.

Case presentation

A 69-year-old woman with fever and persistent involuntary shaking of the perioral area and limbs was diagnosed with SFTS with CNS symptom onset after metagenomic next-generation sequencing (mNGS) of cerebrospinal fluid (CSF) and peripheral blood identified SFTSV. The patient developed a cytokine storm and MOF during the course of the disease, and after aggressive antiviral, glucocorticoid, and gamma globulin treatments, her clinical symptoms improved, her laboratory indices returned to normal, and she had a good prognosis.

This case gives us great insight that when patients with CNS symptoms similar to those of viral encephalitis combined with thrombocytopenia and leukopenia are encountered in the clinic, it is necessary to consider the possibility of SFTS involving the CNS. Testing for SFTSV nucleic acid in CSF and blood (mNGS or polymerase chain reaction (PCR)) should be carried out, especially in critically ill patients, and treatment should be given accordingly.

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Severe fever with thrombocytopenia syndrome (SFTS) is a natural focal disease transmitted mainly by tick bites, and the causative agent is a novel Bunyavirus, also known as SFTS virus (SFTSV), belonging to the Phenuiviridae family and the Bandavirus genus, which was first isolated from patient serum by the Chinese Centre for Disease Control and Prevention in 2010 [ 1 ]. The main features of SFTS include fever, thrombocytopenia, leukopenia and gastrointestinal symptoms, and in severe cases, patients may present with multiple‑organ failure (MOF) symptoms such as shock, respiratory failure, disseminated intravascular coagulation (DIC) and death, with a mortality rate of 5–30% in East Asia [ 2 , 3 ]. SFTS may also present with central nervous system (CNS) involvement, which can severely affect the patient’s disease progression and prognosis and is manifested by seizures, psychiatric symptoms, cognitive impairment, and disorders of consciousness [ 4 , 5 ]. However, reports of patients who present with CNS symptoms as the first symptom and with marked persistent involuntary shaking of the perioral area and limbs are rare.

A 69-year-old female patient was admitted to the hospital with fever for 4 days, involuntary shaking around the mouth and limbs for 3 days, and mental abnormalities for 1 day. The patient was admitted to the emergency department of another hospital 4 days before admission because of fever, where her body temperature reached 38.7 °C and she showed poor mental status, less talking, a loss of appetite, but no headache, vomiting, and limb twitching. A routine blood examination showed a white blood cell (WBC) count of 2.28 × 10 9 /L and a platelet count of 165 × 10 9 /L. When given a cooling infusion for symptomatic treatment, her body temperature would temporarily return to normal. Three days before admission, she experienced persistent involuntary trembling around the mouth and lips, as well as trembling of the tongue and extremities. The trembling of the lips, mouth, and both distal upper limbs was especially bothersome and was aggravated by emotional excitement and accompanied by slurred speech. Two days before admission, she had persistent fever, with a body temperature up to 39.6 °C, and the effect of antipyretic drugs was not good. A routine blood examination performed in another hospital showed a WBC count of 1.78 × 10 9 /L and a platelet count of 81 × 10 9 /L, which was significantly decreased compared with the count from the previous examination. One day prior to admission, the patient experienced babbling, restlessness, irritability, and a decline in time and place orientation and calculation power.

The patient had a many-year history of hypertension, diabetes mellitus and hyperlipidaemia; denied a history of working and living in hilly, forested and mountainous areas and travelling; denied a recent history of mosquito bites; and reported a history of close contact with a pet dog in the last month.

Neurological examination after admission showed that the patient had normal arousal but had unclear speech, hyperactivity, irritability. Her time and place orientation and calculation power decreased. The patient was uncooperative in the pharyngeal reflex examination, and involuntary tongue twitching could be seen when the tongue was stretched out. The remaining cranial nerve examination did not show any abnormalities. Perioral and limb involuntary shaking was obvious and persistent, especially in the perioral area and distal part of both upper limbs. Bilateral tendon reflexes were symmetrical, bilateral pathological signs were negative, and meningeal irritation signs were negative.

On admission, viral encephalitis was considered, and intravenous acyclovir antiviral therapy (0.5 g, q8h) was empirically administered. A comprehensive examination revealed that the patient had MOF: (1) Her platelet count further decreased to 63 × 10 9 /L (normal: 100–300 × 10 9 /L), toxic granules were seen in some granulocytes of the peripheral blood smear, and heterogeneous lymphocytes accounted for 21% of the total. (2) She had impaired liver function with elevated liver enzymes (alanine aminotransferase (ALT), 76 IU/L (normal: 5–40 IU/L); aspartate aminotransferase (AST), 188 IU/L (normal: 8–40 IU/L); and gamma-glutamyl transpeptidase (γ-GT), 177 IU/L (normal: 7–50 IU/L)), which was treated with magnesium isoglycyrrhizinate injection and vitamin C for liver protection. (3) She had acute myocardial injury, with an increased heart rate of > 120 beats/minute and markedly elevated myocardial enzyme and B-type natriuretic peptide levels (myoglobin, 299 ng/mL (normal: 25–58 ng/mL); troponin T, 209 ng/L (normal: 0–14 ng/L); and B-type natriuretic peptide, 9,355 pg/mL (normal: 0-125 pg/mL)). Electrocardiograms (ECGs) showed various atypical manifestations, such as short PR intervals; atrial premature, mild ST-segment depression in leads V2V3; and T-wave changes in multiple leads. Cardiac ultrasound showed a normal left ventricular ejection fraction but abnormal segmental motion of the left ventricular wall, biventricular diastolic insufficiency and a small amount of pericardial effusion. Coenzyme Q10 and trimetazidine were given to improve myocardial energy metabolism, and fluid intake and output were closely monitored. (4) The patient had a bacterial infection of the lungs, combined with type I respiratory failure, which were treated with tracheal intubation and mechanical ventilation immediately to assist respiration and antibiotic antimicrobial therapy. The patient did not have prolonged hypoxic injury. (5) She had impaired renal function, with elevated blood urea nitrogen (BUN) (17.33 mmol/L) (normal: 1.7–8.3 mmol/L) and urinary protein. We administered measures to ensure fluid intake and without the use of nephrotoxic drugs. (6) She had impaired pancreatic function, with elevated lipase (56.5 U/L) (normal: 5.6–51.3 U/L); we administered acid-suppressing drugs to inhibit pancreatic secretion and reduce the load and damage to pancreatic tissue. (7) She had abnormal coagulation, with a prolonged prothrombin time (PT) and thrombin time (TT) (15.7 s (normal: 11–15 s) for PT and 22.6 s (normal: 14–21 s) for TT), decreased fibrinogen (1.8 g/L) (normal: 2–4 g/L), and markedly elevated plasma D-dimer (9.01 µg/mL) (normal: 0.01–0.5 µg/mL) and fibrinogen degradation products (FDPs) (28.36 µg/mL) (normal: 0–5 µg/mL). (8) A thrombus had formed in her right peroneal vein and the intermuscular veins of the right and left calves, for which low molecular heparin anticoagulation was given. (9) Her muscle enzyme profiles were variably elevated (creatine kinase (CK), 335 IU/L (normal: 24–195 IU/L); lactate dehydrogenase (LDH), 1347 IU/L (normal: 109–245 IU/L); and alpha-hydroxybutyrate dehydrogenase (α-HBDH), 645 IU/L (normal: 72–182 IU/L)), correlating with inflammatory response-mediated organ damage. (10) The patient experienced a cytokine storm, with significantly increased inflammatory factors (ferritin > 1500 ng/mL (normal: 11-306.8 ng/mL), interleukin (IL)-6 = 49.88 pg/mL (normal: 0–20 pg/mL), IL-8 = 45.99 pg/mL (normal: 0-21.4 pg/mL), and IL-10 = 25.67 pg/mL (normal: 0-5.9 pg/mL), interferon (IFN)-α = 9.76 pg/mL (normal: 0-7.9 pg/mL), and IFN-γ = 18.7 pg/mL (normal: 0-17.3 pg/mL)) in serum (Table  1 ). (11) Finally, the patient showed an electrolyte balance disorder, as evidenced by hypernatremia (154 mmol/L) (normal: 135–145 mmol/L), hyperchloremia (119 mmol/L) (normal: 96–108 mmol/L), hypocalcaemia (1.92 mmol/L) (normal: 2.03–2.67 mmol/L), and hypophosphatemia (0.54 mmol/L) (normal: 0.84–1.65 mmol/L), and treatments included calcium supplementation, phosphorus supplementation, nasal administration of plain water, and a reduction of sodium and chlorine intake.

Lumbar puncture was performed on the second day after admission (Table  2 ). Cerebrospinal fluid (CSF) was colourless and clear, with a pressure of 190 mmH 2 O (normal: 80–180 mmH 2 O) and a WBC count of 3 × 10 6 /L. CSF cytology showed scattered lymphocytes and a few mononuclear cells. The glucose level and protein counts were normal, chloride was slightly elevated (134 mmol/L) (normal: 118–128 mmol/L), immunoglobulins (Ig) were slightly elevated (IgA, 1.03 mg/dL (normal: 0-0.2 mg/dL); IgM, 0.22 mg/dL (normal: 0-0.2 mg/dL); and IgG, 6.68 mg/dL (normal: 0.48–5.86 mg/dL)), and CSF cytokine levels of IL-6 (27.46 pg/mL) (normal: 0–20 pg/mL) and IL-8 (546.93 pg/mL) (normal: 0-21.4 pg/mL) were elevated. CSF was negative for an autoimmune encephalitis antibody profile (NMDAR, CASPR2, AMPAR1, AMPAR2, LGI1, GABABR, DPPX, and IgLON5), neuroparaneoplastic syndrome antibody profile (Hu, Ri, Yo, CV2, Amphiphysin, GAD65, PNMA2, Recoverin, SOX1, Titin, Tr, and Zic4), and CNS demyelination antibody profile (AQP4, GFAP, MBP, and MOG). Metagenomic next-generation sequencing (mNGS) showed that the number of sequences of a novel Bunyavirus of the Bandavirus genus was 59 in the blood and 12 in the CSF. We also excluded acute febrile illnesses by serum and CSF mNGS, such as dengue fever, chikungunya fever, EB virus infection, renal syndrome hemorrhagic fever, and rickettsial disease.

A diagnosis of SFTS that started with symptoms of CNS and encephalitis due to a novel Bunyavirus was considered based on the patient’s clinical presentation and laboratory test results. With immediate effect, acyclovir was adjusted to the broad-spectrum antiviral drug Foscarnet sodium (3 g, q8h); intravenous infusion of dexamethasone (10 mg qd for five days) and intravenous immunoglobulin (IVIG) (0.4 g/kg for five days) were administered to regulate immune function and inhibit the cytokine storm; nifedipine and benidipine hydrochloride were given to reduce the viral-induced calcium inflow to inhibit viral replication, reduce the viral load and increase the platelet count; clonazepam (1 mg, q8h) was given to relieve the patient’s obvious symptoms of involuntary shaking; and adequate symptomatic supportive therapy was given to ensure adequate calorie and protein intake and to maintain water, electrolyte, blood glucose and acid‒base balance.

After 3 days of hospitalization, the patient’s platelet and WBC counts began to rise gradually and returned to normal levels. After 5 days of hospitalization, the patient’s involuntary shaking and psychiatric symptoms were less severe than before, but compliance with activities was still poor, and her cognitive level still had not returned to normal. After 11 days of hospitalization, the lung infection was better than before, and ventilator withdrawal training was started. After 12 days of hospitalization, cranial magnetic resonance imaging (MRI) was performed, which showed slightly high signals in the bilateral anterior temporal lobe, temporal lobe hook gyrus, insular cortex, and bilateral thalamus on fluid attenuated inversion recovery (FLAIR) and diffusion weighted imaging (DWI) (Fig.  1 a-f). After 13 days of hospitalization, a blood sample was negative for novel Bunyavirus nucleic acid. After 16 days of hospitalization, her condition was significantly better than before, she could perform activities as instructed and answer questions correctly, her time and place orientation returned to normal, and her cognitive level was better than before. A electroencephalogram (EEG) was performed, and a full-lead low-wave amplitude state was observed (Fig.  2 ). After 17 days of hospitalization, the ventilator was completely withdrawn, and the tracheal tube was removed. A repeat lumbar puncture 3 weeks after hospitalization showed a pressure of 110 mmH 2 O, a WBC count of 4 × 10 6 /L, a normal protein count, a slightly elevated glucose level (5.19 mmol/L, compared with a glucose of 7.9 mmol/L over the same period), a slightly elevated chlorine level (130 mmol/L), and a return of Ig to normal. The levels of cytokines IL-6 (4.35 pg/mL) and IL-8 (96.17 pg/mL) decreased significantly compared with the previous levels, and the levels of whole-blood cytokines returned to the normal range (IL-6, 12.22 pg/mL; IL-8, 4.62 pg/mL; IL-10, 1.27 pg/mL; IFN-α, 0 pg/mL; and IFN-γ, 1.14 pg/mL) in serum (Table  1 ). No further novel Bunyaviruses were detected by mNGS of the CSF. Meanwhile, MOF gradually recovered, and liver, heart, lung, kidney, pancreas and coagulation function; the muscle enzyme profile; inflammatory factors; and electrolyte levels gradually returned to normal levels.

After antiviral therapy, immunotherapy, life support and symptomatic treatment, the patient’s vital signs were stable 3 weeks after admission, with clear speech and normal higher cortical function to perform tasks correctly on command. The muscle strength of all four limbs was grade 5, muscle tone was normal, bilateral tendon reflexes existed symmetrically, an ataxia test was normal, bilateral pathological signs were negative, and meningeal irritation signs were negative. She was discharged from the hospital in 23 days after admission. The patient was followed up 1 month after she was discharged from the hospital and is now back to her normal living conditions, with normal functioning of the higher cortex, the ability to take care of herself, and the ability to perform all of the activities she regularly engages in.

Cranial MRI of the patient 12 days after admission. Bilateral anterior temporal lobe (a and d) , temporal lobe leptomeningeal gyrus (a and d) , insular cortex (b and e) , and bilateral thalamus (c and f) FLAIR and DWI sequences with slightly high signals

Sixteen-lead resting-state EEG of the patient 16 days after admission. Simultaneous display an EEG record in monopolar and bipolar montages. A low-amplitude state can be seen in all leads. (a) monopolar montage EEG, (b) bipolar montage EEG

Discussion and conclusions

SFTS is an infectious disease caused by SFTSV infection. The epidemic period is mainly in May-August, and SFTSV is mainly transmitted by tick bites to humans. In recent years, interpersonal and human-animal transmission has also been found. An epidemiological survey of SFTS found that 48% of the patients had had close contact with their pets within two weeks of the onset of the disease [ 6 ]. The general population is susceptible, with a higher risk of infection in residents living in areas such as hills, mountains and forests and in people who spend time outdoors. In this case, SFTSV was isolated from blood and CSF. There was no history of tick bites or travel in the wild, but there was a history of close contact with a pet dog within the past month, and we hypothesized that the infected dog might have been the source of SFTSV in this patient.

The pathogenesis of CNS involvement in SFTS patients is unclear. Previous studies have demonstrated that Bunyaviruses have neurological properties of attack, and Park et al. found viral transcripts of novel Bunyaviruses in the brain and spinal cord of an aged model ferret. It is hypothesized that novel Bunyaviruses also involve the CNS, with consequent symptoms [ 7 ]. Possible mechanisms by which SFTSV attacks the CNS include direct invasion, cytokine storms, and impaired immune function. Kaneko et al. [ 8 ] performed an autopsy on a patient with SFTS with rapid CNS involvement, and the pathological findings revealed a massive infiltration of macrophages with high haematoxylin content and inflammatory cells around the microvessels of the cerebral pontine, fibrin deposition in the vessels, and focal degenerative lesions in some neuronal cells. In a variety of brain tissues, positive SFTSV nucleocapsid protein antigens were observed in the immunoblasts infiltrating the vascular lumen, suggesting that SFTSV can invade the CNS directly for disease development. The availability of agents that recognize these antigens also suggest immunoassays are possible and available for serodiagnosis. For example, serum enzyme linked immunosorbent assay or immunofluorescence to determine SFTSV antigens and antibodies have been used for clinical diagnosis [ 9 ]. Several studies [ 10 , 11 , 12 , 13 , 14 , 15 ] have found that the blood levels of several cytokines, including IFN-α, IFN-γ, IL-6, IL-8, IL-10, tumour necrosis factor (TNF)-α, and monocyte chemotactic protein (MCP)-1, are elevated in patients with SFTS, and IL-8 and MCP-1 levels in the CSF are significantly higher than the blood of those who present with CNS symptoms [ 10 ], suggesting that a cytokine storm may increase vascular permeability and prompt SFTSV to cross the blood‒brain barrier (BBB) and invade the CNS. SFTSV was found in the CSF of this patient, suggesting that the virus had invaded the patient’s CNS. The patient’s blood levels of IL-6, IL-8, IL-10, IFN-α, and IFN-γ were markedly elevated compared with normal ranges; IL-6 and IL-8 were elevated in the CSF; and CSF IL-8 levels were significantly higher than the blood levels, which was consistent with the results of a previous study [ 10 , 11 , 12 , 13 , 14 , 15 ], further suggesting that the cytokine storms induced by multiple elevated cytokines may increase BBB vascular permeability and contribute to the SFTSV invasion of the CNS. In patients with SFTS complicated by neurological involvement, protein and glucose levels in the CSF are normal and that an increase in leukocytes in the CSF may be uncommon. However, in the case of a high suspicion both on a clinical and epidemiological level in countries where the infection exists, in these patients the search for MCP-1 and IL-8 in the CSF and serum is indicated and CSF viral RNA detection are recommended.

According to the course of infection, SFTS can be divided into four periods: the incubation period, the febrile period, the MOF period, and the recovery period [ 1 , 2 , 3 , 16 ]. Patients with SFTS can present with neurological symptoms, which usually appear approximately 5 days after the onset of the disease (Table  3 ) and are often regarded as a complication of SFTS, which has been referred to as SFTS-associated encephalopathy/encephalitis (SFTSAE) [ 10 ]. SFTSAE mainly manifests as headache, seizures, mental abnormality, irritability, limb convulsions, cognitive impairment, and impaired consciousness, with an incidence of approximately 19.1-57.02% [ 4 , 5 , 11 , 17 ]. Most patients with SFTSAE develop impaired consciousness, such as coma, before their condition is taken seriously, which leads to a poor prognosis for the patients [ 4 , 18 ]. Most clinicians rely on the clinical manifestations to make the clinical diagnosis. SFTSV has rarely been isolated from CSF. We screened studies and case reports of SFTS with CNS involvement and found no reports of disease onset with CNS symptoms such as marked persistent involuntary shaking of the perioral area and extremities. In this case, the patient first presented with fever, followed by persistent involuntary tremors of the perioral area and limbs and mental behavioural abnormalities such as rambling, irritability and agitation; furthermore, the whole-genome sequence of SFTSV was found by mNGS of blood and CSF. The case reported here is a case of SFTS with CNS symptoms onset, accompanied by perioral and extremity persistent involuntary shaking, which has not been previously reported in the literature. It has been reported in the literature that SFTS patients can have tremors of limbs and muscles [ 8 , 17 , 19 ], but most of them occurred in the middle and late stages of the disease, and the tremor amplitude was small. In this case, the patient had large-amplitude involuntary shaking of the limbs that was persistent and intensified during agitation, which immediately attracted the clinician’s attention. An additional movie file shows this in more detail [see Additional file 1 ]. However, the specific underlying mechanism is not clear, and a description of similar symptoms of viral encephalitis and an analysis of the underlying mechanism have not been found before; therefore, further studies are needed. The course of the disease in this patient was consistent with the general pattern, with the clinical experience of the febrile period, the MOF period, and the recovery period. The febrile period lasted approximately 4 days, followed by MOF involving the liver, heart, lungs, kidneys, and pancreas, and then the recovery period began approximately 2 weeks after the disease onset, with clinical symptoms gradually returning to normal.

There are fewer reports on neurological-related ancillary investigations (CSF, cranial imaging, and EEG) in SFTS patients with CNS involvement, and we analyse this because SFTS patients rarely start with CNS symptoms and go directly to the neurology department and because such patients are generally more severely ill, making it difficult for them to cooperate in completing the relevant investigations. In a few previous studies, lumbar puncture CSF tests in SFTS patients with CNS symptoms were mostly normal, with few abnormal changes in leukocyte counts, sugars and proteins [ 10 , 20 ]. Park et al. [ 10 ] analysed head imaging and EEG in a series of SFTS patients presenting with CNS symptoms, and no new focal lesions were seen on imaging in any of the brain parenchyma, suggesting that the imaging was not specific and that the EEG in the majority of the patients showed a slow-wave background rhythm (δ-θ), a common feature of encephalitis/encephalopathy. In this patient, two lumbar punctures were performed successively, and no CSF leukocyte abnormalities were observed in any of them either; it was presumed that SFTSV infection was less likely to involve the meninges. We performed cranial MRI and EEG on the patient 12 and 16 days after admission, respectively, and slightly high signals were observed in the bilateral anterior temporal lobes, temporal lobe hook gyrus, insular cortex, and bilateral thalamus in the FLAIR and DWI sequences of cranial MRI, all of which were consistent with the general imaging manifestations of viral encephalitis and were presumed to be related to viral invasion. In addition, we should consider the similarities and differences between the above MRI changes and cortical laminar necrosis associated with hypoxia or hypotension. We found that both had MRI high signals distributed along the cortex. However, this patient’s cranial MRI showed cortical high signals only in FLAIR and DWI sequences, and no abnormal signal was found in T1WI, which was the most obvious difference from cortical laminar necrosis. Furthermore, the patient did not show hypotension or significant hypoxic injury, so the changes on cranial MRI were more likely to be inflammatory changes of viral encephalitis and less relevant to cortical laminar necrosis. The background rhythm of the EEG was an α rhythm, and the whole leads were in low amplitude, which was different from previous studies [ 10 ]. It was presumed that the patient’s brain inflammation had tended to recover at that time, but the suppression of cortical function was remained.

There are no specific drugs for the treatment of CNS symptoms in SFTS, and symptomatic supportive treatment is the mainstay. In vitro and ex vivo studies have found that nifedipine or benidipine hydrochloride can inhibit SFTSV replication, reduce viral load, increase platelet counts, and reduce morbidity and mortality, as confirmed in a retrospective clinical study [ 21 , 22 ]. Glucocorticoids can inhibit the cytokine storms caused by the overproduction of cytokines and reduce patient mortality [ 12 , 13 , 23 ], and a Japanese report documented that three SFTS patients with impaired consciousness recovered without any neurological sequelae after short-term glucocorticoid treatment. However, the authors also suggested that the dosage should be minimized and the duration of administration should be shortened to inhibit cytokine storms and provide systemic benefit, rather than high doses or prolonged use, to avoid side effects [ 24 ]. Gamma globulin, which triggers complement activation and viral neutralization and influences the differentiation process of Schwann cells to increase their regenerative potential [ 25 ], has been used to treat other virus-induced encephalitides and can be used for the treatment of CNS symptoms in SFTS. Two successful cases of combined glucocorticoid and IVIG therapy were reported in Korea [ 26 ]. Two case reports documented that plasma exchange therapy reduced cytokine levels but not viral load, presumably making plasma exchange more effective at an early stage [ 27 , 28 ]. However, these are case reports, and the findings should be confirmed by large-scale randomized controlled studies. In this case, the patient was given the broad-spectrum antiviral drug foscarnet sodium, intravenous infusion of dexamethasone and IVIG to regulate the immune function of the body and inhibit the inflammatory storm, nifedipine and benidipine hydrochloride to inhibit viral replication and reduce the viral load, and other symptomatic treatments. The patient’s clinical manifestations and laboratory indicators gradually improved.

The prognosis of patients with SFTS is related to numerous factors, and studies have shown that advanced age; significant elevations in ALT, AST, CK, CK-MB, LDH, γ-GT, and BUN; low platelet count; persistent lowering of blood calcium; and the presence of CNS symptoms are all important influences that can lead to a poor prognosis [ 29 , 30 , 31 , 32 , 33 , 34 ]. Most of these are commonly used to monitor cardiac, hepatic and renal function, and significant abnormalities in their results indicate more severe organ damage and dysfunction. In addition, there is a statistically significant difference in serum viral copy number between deceased and non-dead patients. The mean viral copy number was higher in deceased patients than in surviving patients, and patients with higher copy numbers had higher mortality rates [ 35 , 36 ]. It was shown that the serum viral load detected by polymerase chain reaction (PCR) on admission was higher in SFTSAE patients than in non-encephalitis patients [ 11 ]. The above suggests a relationship between patient serum number of SFTSV RNA copies and encephalitis CNS symptoms and mortality in SFTS patients. CNS symptoms are often considered to be associated with fatal outcomes in patients with SFTS [ 33 ], and early diagnosis and treatment of neurological symptoms can help reduce mortality. Advanced age; long intervals between onset and admission; comorbid diabetes mellitus or subcutaneous haemorrhage; pulmonary rales; low platelet count; elevated neutrophil percentages and LDH, CK, and C-reactive protein (CRP) levels; and decreased chloride concentrations are significantly associated with the development of CNS symptoms and should be taken into consideration in clinical practice [ 11 , 17 ]. We believe that changes in platelet count and CK-MB should be monitored in patients with SFTSAE. As shown in previous, decreased platelet counts and high CK-MB levels are risk factors for poor prognosis in patients with SFTS. The presence of encephalitis is evidence of a more critical condition. Monitoring changes in platelet counts may provide an initial indication of the direction of the patient’s regression. It has been found that in cardiac enzyme profiles, patients presenting with CNS symptoms have elevated CK levels earlier than LDH and AST levels, and elevated liver enzyme levels later than cardiac enzymes [ 17 ]. Therefore, early monitoring of CK-MB levels may have a predictive effect on the development of CNS symptoms in patients. Although the mortality rate of SFTS patients presenting with CNS symptoms is significantly higher [ 11 ], several studies have found [ 11 , 37 , 38 ] that the long-term prognosis of surviving patients is good, with no obvious sequelae after active treatment. In this case, the patient’s laboratory indicators were consistent with the factors leading to a poor prognosis, and the CNS symptoms were prominent, suggesting that the condition was critical, but with timely administration of treatment, the patient’s condition eventually returned to normal.

In summary, we report a case of SFTS in a patient who started with CNS symptoms accompanied by marked persistent involuntary perioral and extremity shaking, and the whole-genome sequence of SFTSV was found by mNGS of both serum and CSF (It is important to note that hospitals where mNGS analysis is unavailable should use real-time fluorescent quantitative PCR to detect SFTS-specific nucleic acids in serum and CSF.). This has given us great insight into the fact that SFTS should be considered a possible cause when patients present with common CNS symptoms of viral encephalitis, such as mental behavioural abnormalities, convulsions, and cognitive deficits, or rare symptoms, such as persistent involuntary shaking of the perioral area and limbs in the rare case of this patient, combined with thrombocytopenia and leukopenia. Prompt lumbar puncture examination for SFTSV should be performed, and appropriate treatment should be given aggressively to reduce mortality.

Data availability

No datasets were generated or analysed during the current study.

Abbreviations

severe fever with thrombocytopenia syndrome

severe fever with thrombocytopenia syndrome virus

multiple-organ failure

disseminated intravascular coagulation

central nervous system

metagenomic next-generation sequencing

cerebrospinal fluid

alanine aminotransferase

aspartate aminotransferase

gamma-glutamyl transpeptidase

electrocardiogram

blood urea nitrogen

prothrombin time

thrombin time

fibrinogen degradation products

creatine kinase

lactate dehydrogenase

alpha-hydroxybutyrate dehydrogenase

interleukin

immunoglobulin

intravenous immunoglobulin

magnetic resonance imaging

fluid attenuated inversion recovery

diffusion weighted imaging

electroencephalogram

tumour necrosis factor

monocyte chemotactic protein

blood-brain barrier

severe fever with thrombocytopenia syndrome -associated encephalopathy/ encephalitis

polymerase chain reaction

C-reactive protein

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This project was supported by National Key Research and Development Program of China (2020YFC2005403), and by China Association for Promotion of Health Science and Technology (JKHY2023001).

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Dawei Shan and Yan Zhang contributed to the conception and design of the manuscript. Dawei Shan collected the data and drafted the manuscript. Yan Zhang, Weibi Chen, Gang Liu, Huimin Zhang and Shuting Chai reviewed and modified the manuscript. All authors contributed to manuscript revision and read and approved the final submitted version.

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12883_2024_3664_MOESM1_ESM.mp4

Supplementary Material 1. File name: Additional file 1. File format: mp4. Title of data: Video of patient with persistent involuntary shaking of the perioral area and limbs. Description of data: We took this video on day 2 after the patient was admitted to the hospital. The patient develops persistent involuntary shaking of the perioral area and limbs, especially in the perioral area and distal limbs, which is aggravated by agitation and is accompanied by slurred speech.

Supplementary Material 2. CARE Checklist of information to include when writing this case report.

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Shan, D., Chen, W., Liu, G. et al. Severe fever with thrombocytopenia syndrome with central nervous system symptom onset: a case report and literature review. BMC Neurol 24 , 158 (2024). https://doi.org/10.1186/s12883-024-03664-6

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Motor polyradiculoneuropathy as an unusual presentation of neurobrucellosis: a case report and literature review

• Ahmad Alikhani 1 ,
• Noushin Ahmadi 1 ,
• Mehran Frouzanian 2 &
• Amirsaleh Abdollahi 2  

BMC Infectious Diseases volume  24 , Article number:  491 ( 2024 ) Cite this article

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Brucellosis, a zoonotic disease caused by Brucella species, poses a significant global health concern. Among its diverse clinical manifestations, neurobrucellosis remains an infrequent yet debilitating complication. Here, we present a rare case of neurobrucellosis with unusual presentations in a 45-year-old woman. The patient’s clinical course included progressive lower extremity weakness, muscle wasting, and double vision, prompting a comprehensive diagnostic evaluation. Notable findings included polyneuropathy, elevated brucella agglutination titers in both cerebrospinal fluid and blood, abnormal EMG-NCV tests, and resolving symptoms with antibiotic therapy. The clinical presentation, diagnostic challenges, and differentiation from other neurological conditions are discussed. This case underscores the importance of considering neurobrucellosis in regions where brucellosis is prevalent and highlights this rare neurological complication’s distinctive clinical and radiological features. Early recognition and appropriate treatment are crucial to mitigate the significant morbidity associated with neurobrucellosis.

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Introduction

Brucellosis, caused by Brucella species, is an infectious ailment recognized by various names such as remitting, undulant, Mediterranean, Maltese, Crimean, and goat fever. Humans contract it through the consumption of unpasteurized milk and dairy products, undercooked meat, or skin contact with infected livestock [ 1 , 2 , 3 ]. Various Brucella species, including Brucella melitensis (primarily sourced from sheep and goats), Brucella abortus (found in cattle), Brucella suis (associated with pigs/hogs), and Brucella canis (linked to dogs), can lead to illness in humans [ 3 , 4 , 5 ]. While brucellosis in humans is rarely fatal, it can lead to disability [ 6 ]. Brucellosis ranks among the most prevalent zoonotic diseases, impacting approximately 500,000 individuals yearly [ 7 ]. The combined estimate for the prevalence of brucellosis was 15.53% [ 8 ].

Neurobrucellosis, a rare complication of systemic brucellosis, can occur in adult and pediatric cases [ 9 ], and can manifest at any stage of the disease. They can present in various clinical presentations such as meningitis, encephalitis, meningoencephalitis, myelitis, radiculopathy, polyneuropathy, stroke, cerebral venous thrombosis, and occasionally psychiatric symptoms [ 10 , 11 ]. Although the mortality rate is low, patients often experience persistent neurological issues following neurobrucellosis [ 12 ]. Studies suggest that around 20% of neurobrucellosis cases result in lasting neurological problems [ 13 ]. It is uncommonly considered in cases of meningoencephalitis or polyneuropathy, making it crucial for clinicians to have a high suspicion of it in patients displaying such symptoms, especially in endemic regions, to prevent severe clinical outcomes. In this study, we present a rare case of neurobrucellosis with unusual clinical presentations in a patient admitted to our center.

Case presentation

A 45-year-old female patient, with no prior medical history, presented to our center after enduring distal pain and weakness in her lower extremities for approximately 10 months. Over this period, the muscle weakness progressed, affecting proximal muscles of upper and lower limbs, and leading to a substantial weight loss of 25–30 kg despite maintaining appetite. Initially dismissive of the limb weakness and pain, the patient sought medical attention six months after symptom onset due to the worsening symptoms and gait impairment. Over the subsequent four months, she underwent multiple medical evaluations and tests, including a lumbar X-ray. Following these initial investigations and due to low serum vitamin D levels, vitamin D and calcium supplements were prescribed, and lumbar MRI were requested for further evaluation. (Table  1 )

Upon referral to an infectious disease specialist, the patient’s history of local dairy consumption and positive serologic test for brucellosis prompted treatment with rifampin and doxycycline. However, the patient’s condition deteriorated significantly five days after starting this treatment. She experienced severe gait disorder, lower extremity weakness, diplopia, and blurred vision that had gradually worsened over two weeks. Subsequently, she presented to our center for further assessment.

Upon admission, the patient was unable to stand even with assistance and exhibited diplopia. Cranial nerve examination revealed no abnormalities, except for the II, III, and IV cranial nerves, which could not be thoroughly examined due to the presence of diplopia. The patient tested negative for Kernig and Brudzinski signs. There were no palpable supraclavicular or inguinal lymph nodes. Physical examinations of the breast, axilla, lungs, heart, and abdomen were unremarkable. Muscle strength was reduced in the lower extremities, and deep tendon reflexes of the knee and Achilles were absent. The plantar reflex was non-responsive, and certain reflexes, including biceps, triceps, and brachioradialis, were absent despite normal movement of the upper extremities. Anorectal muscle tone and anal reflex were normal.

Further investigations included normal urinalysis and abdominal and pelvic ultrasound. Chest X-ray and brain CT were also ordered. Due to the patient’s refusal of lumbar puncture, a suspicion of neurobrucellosis led to the initiation of a three-drug regimen (Table  2 ); ceftriaxone 2 g IV twice daily, rifampin 600 mg PO daily, and doxycycline 100 mg PO twice daily. The ophthalmology consultation did not reveal any ocular pathology, and the neurologist ordered brain MRI and EMG-NCV tests. The patient’s brain MRI was unremarkable, but EMG-NCV showed sensory and motor polyneuropathy. Consequently, intravenous immunoglobulin (IVIG) therapy was initiated at a daily dose of 25 g. After five days, the patient consented to lumbar puncture, confirming the diagnosis of brucellosis. Co-trimoxazole 960 mg PO three times daily was added to her treatment regimen, and IVIG therapy continued for seven days. Following a 3-day course of IVIG treatment, the neuropathy symptoms showed significant improvement. By the seventh day, there was a notable enhancement in limb strength, particularly in the upper limbs, reaching a 2-point improvement. After undergoing three weeks of intravenous therapy, the patient transitioned to oral medication. Despite disagreement regarding the necessity of a second CSF examination, the patient was discharged with a prescription for doxycycline, rifampin, and cotrimoxazole. Upon discharge, the patient could walk with the aid of a walker. However, within a month, a slight limp persisted, and by the third-month post-discharge, all symptoms had resolved completely.

Brucellosis is widely spread globally, with more than half a million reported human cases annually [ 14 , 15 ]. Countries like Kenya, Yemen, Syria, Greece, and Eritrea have experienced high rates of brucellosis. The situation of brucellosis has shown signs of improvement in many epidemic regions. However, new areas with high occurrences of this disease continue to emerge, particularly in Africa and the Middle East, where the incidence of the disease varies [ 16 ]. Brucellosis is linked to various neurological complications collectively known as neurobrucellosis, which is an uncommon condition, and only a few cases have been reported globally [ 17 , 18 , 19 , 20 , 21 ]. Our patient exhibited muscle weakness, polyneuropathy, and inability to walk, which are often not regarded as indicative of a brucella infection by many physicians. While the diagnosis of neurobrucellosis can typically be confirmed through classical clinical signs, radiological examinations, and serological tests, patients might not always display typical symptoms, as observed in our case. Hence, in regions where the disease is prevalent, clinicians should maintain a high level of suspicion if patients do not show improvement with standard treatment. Additionally, the lack of awareness among healthcare professionals and limited access to advanced laboratory facilities can lead to misdiagnosis.

The frequent manifestations of neurobrucellosis include meningitis or meningoencephalitis. Typically, it starts with a sudden headache, vomiting, and altered mental state, which can progress to unconsciousness, with or without seizures [ 22 ]. Additionally, brucellosis can lead to several central nervous system issues such as inflammation of cerebral blood vessels, abscesses in the brain or epidural space, strokes, and cerebellar ataxia. Peripheral nerve problems may include nerve damage or radiculopathy, Guillain-Barré syndrome, and a syndrome resembling poliomyelitis [ 13 ]. Nevertheless, the patient exhibited no indications of seizures, brain hemorrhage, stroke, or focal neurological impairments. Instead, the observed symptoms were consistent with radiculopathy and muscular weakness.

In only 7% of neurobrucellosis cases, the peripheral nervous system is affected. Remarkably, our case falls within this rare category, adding to its unique and intriguing nature. Previous case studies have detailed polyradiculoneuropathies, manifesting as acute, subacute, or chronic forms [ 23 ]. Our patient’s condition aligns with chronic motor polyradiculopathy. Interestingly, some of these cases exhibit sensory deficits or resemble Guillain-Barré syndrome [ 23 , 24 ]. In a prior case study conducted by Abuzinadah and colleagues, a comparable case was described as a subacute motor polyradiculopathy. The patient exhibited gradual bilateral lower limb weakness over three weeks, eventually leading to loss of mobility within seven weeks. Brucella was isolated from the cerebrospinal fluid after a two-week incubation period, and high antibody titers were detected in the patient’s serum [ 23 ]. In another study led by Alanazi and colleagues, a 56-year-old man initially diagnosed with Guillain-Barré syndrome experienced worsening symptoms despite appropriate treatment. Following plasma exchange and antibiotics, his condition improved temporarily, only to relapse, raising suspicion of chronic inflammatory demyelinating polyneuropathy, and treatment with IVIG resulted in substantial improvement. Upon further investigation, he was diagnosed with brucellosis [ 24 ]. This highlights the importance of recognizing GBS-like symptoms in regions where brucellosis is prevalent, prompting clinicians to consider the possibility of brucellosis in their diagnosis.

While there are no established criteria for diagnosing neurobrucellosis [ 25 ], certain articles have suggested several methods for its diagnosis. These methods include the presence of symptoms aligning with neurobrucellosis, isolating brucella from cerebrospinal fluid (CSF) or detecting a positive brucella agglutination titer in CSF, observing lymphocytosis, elevated protein, and decreased glucose levels in CSF, or identifying specific diagnostic indicators in cranial imaging such as magnetic resonance imaging or computed tomography (MRI or CT) [ 13 , 26 , 27 , 28 ]. Neurobrucellosis does not present a distinct clinical profile or specific CSF characteristics. Imaging observations of neurobrucellosis fall into four categories: normal, inflammatory (indicated by granulomas and enhanced meninges, perivascular spaces, or lumbar nerve roots), alterations in white matter, and vascular changes [ 29 ]. We suspected neurobrucellosis based on the patient’s clinical symptoms, geographic correlation, high brucella agglutination test titers in both cerebrospinal fluid and blood, symptom resolution following treatment, and the exclusion of other common causes.

In Iran, one differential diagnosis often confused with brucellosis is tuberculosis, as both chronic granulomatous infectious diseases are prevalent here [ 30 , 31 ]. Neurobrucellosis and tuberculosis exhibit significant similarities in clinical symptoms, lab results, and neuroimaging findings. However, deep grey matter involvement and widespread white matter lesions seen in neuroimaging, resembling demyelinating disorders, appear to be distinctive to brucellosis [ 32 ]. There is a noticeable similarity in the clinical symptoms and laboratory findings of brucellosis and tuberculosis [ 33 ]. It is crucial to thoroughly eliminate the possibility of tuberculosis in any suspected or confirmed brucellosis cases before starting antibiotic treatment.

Due to the challenging nature of treating brucellosis and the likelihood of experiencing relapses, it is crucial to provide an extended course of treatment [ 27 ]. This treatment approach should involve a combination of antibiotics that can easily penetrate the cell wall and effectively reach the central nervous system [ 27 , 34 ]. Neurobrucellosis is treated with 3 to 6 months of combination therapy comprising doxycycline, rifampicin, and ceftriaxone or trimethoprim-sulfamethoxazole [ 35 ], similar to the treatment administered to our patient. For patients allergic to cephalosporins, quinolones are recommended, which are considered to be effective in treating brucellosis [ 36 , 37 ]. In complicated situations such as meningitis or endocarditis, streptomycin or gentamicin is administered in the initial 14 days of treatment, in addition to the previously mentioned regimen. Timely and proper treatment results in a positive prognosis, with a less than 1% fatality rate for such complex cases [ 17 , 38 ]. Our patient experienced a highly positive outcome following the prescribed therapy. Initially relying on a walker, a slight limp endured for a month, and by the third month after discharge, all symptoms completely disappeared.

The present study underscores the significance of considering neurobrucellosis as a potential diagnosis when evaluating muscle weakness and radiculopathy, especially in regions where the disease is prevalent. A comprehensive patient history, precise clinical examination, positive serology in blood or cerebrospinal fluid, imaging results, or cerebrospinal fluid analysis can contribute to establishing a conclusive diagnosis.

Data availability

The datasets generated and/or analysed during the current study are not publicly available due to our team’s privacy concerns but are available from the corresponding author on reasonable request.

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A.A oversaw and treated the case, including the entire revision process. N.A. contributed to the article’s composition. M.F. authored the discussion section, along with the complete revision. AS.A. played a role in crafting the case report discussion and participated in the entire revision process.

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Alikhani, A., Ahmadi, N., Frouzanian, M. et al. Motor polyradiculoneuropathy as an unusual presentation of neurobrucellosis: a case report and literature review. BMC Infect Dis 24 , 491 (2024). https://doi.org/10.1186/s12879-024-09365-2

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Received : 04 December 2023

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DOI : https://doi.org/10.1186/s12879-024-09365-2

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• Neurobrucellosis
• EMG-NCV tests
• Polyradiculoneuropathy
• Antibiotic therapy
• Intravenous immunoglobulin therapy
• Zoonotic disease
• Gait disorder
• Lower extremity weakness
• Blurred vision

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