literature review and its importance pdf

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

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

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

What are Literature Reviews?

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

Goals of Literature Reviews

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

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

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

What kinds of sources require a Literature Review?

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

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

Types of Literature Reviews

What kinds of literature reviews are written?

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

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

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

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

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

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

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

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

Literature Reviews in the Health Sciences

• UConn Health subject guide on systematic reviews Explanation of the different review types used in health sciences literature as well as tools to help you find the right review type
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Importance of Literature Review

research proposal as a problem to investigate, it usually has to be fairly narrow and focused, and because of this it can be difficult to appreciate how one's research subject is connected to other related areas. Therefore, the overall purpose of a literature review is to demonstrate this, and to help the reader to understand how your study fits into a broader context. This paper seeks to examine this topic of literature review, its significance and role in research proposal and report. It will start by explaining in detail what literature is; by citation of different scholars and its constituent components, such as the theoretical framework. Thereafter, it will look at the importance of literature review and its role in research proposals and reports. Finally, a conclusion will be written based on this topic. A Literature Review is a critical review of existing knowledge on areas such as theories, critiques, methodologies, research findings, assessment and evaluations on a particular topic. A literature review involves a critical evaluation identifying similarities and differences between existing literatures and the work being undertaken. It reviews what have already been done in the context of a topic. Therefore, on the basis of the existing knowledge, people can build up innovative idea and concept for further research purpose (Cooper, 1998). In doing empirical literature review is reading reports of other relevant studies conducted by different researchers. In doing so, a researcher gets knowledge and experiences that were established by other researchers when conducting their studies. While Conceptual framework is a set of coherent ideas or concepts organised in a manner that makes them easy to communicate to others. It represents less formal structure and used for specific concepts and propositions derived from empirical observation and intuition (ibid). According Aveyard, H. (2010) Theoretical framework is a theoretical perspective. It can be simply a theory, but can also be more general a basic approach to understanding something. Typically, a theoretical framework consists of concepts, together with their definitions, and existing framework must demonstrate an understanding of theories, and existing framework demonstrate an understanding of theories and concepts that are relevant to the topic of your research proposal and that will relate it to the broader fields of knowledge in the class you are taking.

Related Papers

Journal of African Interdisciplinary Studies

Ronald Mensah

This article critically discusses, the relationship between conceptual framework and theoretical framework drawing on their differences and similarities. The article has made it very clear that whereas the theoretical framework is drawn from the existing theoretical literature that you review about your research topic, a conceptual framework is a much broader concept that encompasses practically all aspects of your research. The latter refers to the entire conceptualisation of your research project. It is the big picture, or vision, comprising the totality of research. Methodologically, the paper used systematic and experiential literature review to draw supporting scholarly literature by authorities in the field and made inferences, sound reasonings and logical deductions from these authorities. The primary aim of this paper is to help researchers and students to understand the convergence and the divergence of theoretical and conceptual frameworks in order to appropriately be applied in research and academic writing discourses. Understanding the conceptual framework affects research in many ways. For instance, it assists the researcher in identifying and constructing his/her worldview on the phenomenon to be investigated. Also, it is the simplest way through which a researcher presents his/her asserted remedies to the milieu he/she has created. In addition, this accentuates the reasons why a research topic is worth studying, the assumptions of a researcher, the scholars he/she agrees with and disagrees with and how he/she conceptually grounds his/her approach. Paying attention, to the theoretical framework and its impact on research, it can be mentioned that theoretical framework provides a structure for what to look for in the data, for how you think of how what you see in the data fits together, and helps you to discuss your findings more clearly, in light of what existing theories say. It helps the researcher to make connections between the abstract and concrete elements observed in the data. In conclusion, both theoretical framework and conceptual framework are good variables which are used to inform a study to arrive at logical findings and conclusions. It is therefore recommended by researchers that; a good theoretical framework should be capable of informing the concepts in a research work.

Abey Dubale

Martin Otundo Richard

ABSTRACT A number of researchers either in scientific, social or academic researchers have found it difficult to differentiate between the theoretical and conceptual framework and their importance. This is a simple overview of the basic differences and some similarities between the theoretical and conceptual framework that is aimed at helping the learners and other researchers get a fast grasp of what can help them use the two effectively in their studies. A summary has been provided at the last part of the document that can aid one get the required information in his or her research.

Tonette S Rocco , Maria Plakhotnik

Abstract This essay starts with a discussion of the literature review, theoretical framework, and conceptual framework as components of a manuscript. This discussion includes similarities and distinctions among these components and their relation to other sections of a manuscript such as the problem statement, discussion, and implications. The essay concludes with an overview of the literature review, theoretical framework, and conceptual framework as separate types of manuscripts.

Ewnetu Tamene

The impetus of this paper is the irreplaceable role yet confusing use of the term " conceptual framework " in research literature. Even though, there is a consensus among scholars of various field of study that conceptual framework is essential element of research endeavors, yet it is used interchangeably with theoretical framework, that create confusion. As a PhD student on the pre-proposal work, this makes me more anxious. Then my intent is to explore its conceptual meaning and purposes by bringing together similar meanings from different scholars with a view to shed some light on its understanding and its use in research. Hence, in attempting to address this, the following key terms; Concept, conceptual framework research design and theoretical framework are defined briefly as to help decipher the conceptual ties among them and illuminate the conceptual meaning and purpose of conceptual framework. The schematic representation of conceptual framework is developed based on the conceptual meaning provided by scholars. In doing this it is attempted to show conceptual meaning of conceptual framework in relation to research design, paradigms and philosophical assumptions that delineate it from theoretical framework. Conceptual framework serves essential role in inductive research design, while theoretical framework serves similar role in deductive research design.

This is an opinion piece on the subject of whether or not 'theoretical' and 'conceptual' frameworks are conceptual synonyms, or they refer to different constructs. Although, generally, a lot of liter ature uses these two terms interchangeably – suggesting that they are conceptually equivalent, the researcher argues that these are two different constructs – both by definition and as actualised during the research process. Thus, in this paper, the researcher starts by developing his argument by examining the role of theory in research, and then draws a distinction between areas of research that typically follow deductive versus inductive approaches, with regard to both the review of literature and data collection. The researcher then subsequently argues that whereas a deductive approach to literature review typically makes use of theories and theoretical frameworks, the induct ive approach tends to lead to the development of a conceptual framework – which may take the form of a (conceptua l) model. Examples depicting this distinction are advanced.

Nauka i društvo

Darko M Marković

Conceptual framework is the foundation of scientific research, and it is formed from previous knowledge about the researched phenomenon. It is an integral part of theoretical framework, made with the aim to include key terms, presented within the bibliography, and create a suitable platform to develop the research correctly. More often with students preparing their final papers, and not so rarely with established researchers, the problem arises when it comes to conceptualizing research. A common unknown is how to divide conceptual from theoretical framework, and what conceptual framework of scientific research actually is. The aim of this paper is to clarify conceptualization of scientific research, and, by clarifying it, to point out the significance of differentiation between conceptual and theoretical framework, going on to give basic guidelines on how to form one, and therefore ease the understanding of it and application in scientific research.

Aliya Ahmed

Elock E Shikalepo

Conducting educational research involves various stages, with an interdependence and inter-relationship which can be both iterative and progressive in nature. One of these stages is the review of literature sources related to the focus of the research. Reviewing related literature involves tracing, examining, critiquing, evaluating and eventually recommending various forms of contents to the intents of the research based on the content’s typicality, relevance, correctness and appropriateness to what the research intends to achieve. The main variables as stated in the title of the research, the research questions, the research objectives and the hypotheses, dictates the literature sources to review. Reviewing literature focuses on the existing related topics that bear relevance to the title of the research and through which reviewing, appropriate theories can be picked up as the review of related topics and phrases goes on. As soon as the related topics are reviewed and main points noted, the reviewing process proceeds to review the theories underpinning the study. Some of these theories would have been established while reviewing the related topics and can now gain momentum, while other theories can now be generated considering the title, research questions, research objectives and findings of the topics reviewed and discussed earlier. Reviewing related topics generates main points of arguments, solutions, gaps and propositions. Similarly, reviewing theories does produce the same set of corresponding or contrasting agreements, gaps and propositions. Despite reviewing different sources of literature, it is the same research at hand, with same objectives and same methodological layout. Hence, a need to shape a strategic, literature direction for the research by consolidating the key findings of the different sources reviewed, in view of the intents of the research. The process of consolidating the multiplicity of key literature findings relevant to the research into a whole single unit, with one standpoint revealing the strategic literature direction for the research, is called constructing a conceptual framework. The end product of this construction is the conceptual framework, which is the informed and consolidated results presented narratively or schematically, revealing the strategic position of the study in relation to what exists in literature.

pinky adona

Many students, both in the undergraduate and graduate levels, have difficulty discriminating the theoretical from the conceptual framework. This requires a good understanding of both frameworks in order to conduct a good investigation. This article explains the two concepts in easily understandable language. Read on to find out. Many graduating college students and even graduate students have difficulty coming up with the conceptual framework and the theoretical framework of their thesis, a required section in thesis writing that serves as the students' map on their first venture into research. The conceptual framework is almost always confused with the theoretical framework of the study. What is the difference between the conceptual and the theoretical framework? A conceptual framework is the researcher's idea on how the research problem will have to be explored. This is founded on the theoretical framework, which lies on a much broader scale of resolution. The theoretical framework dwells on time tested theories that embody the findings of numerous investigations on how phenomena occur. The theoretical framework provides a general representation of relationships between things in a given phenomenon. The conceptual framework, on the other hand, embodies the specific direction by which the research will have to be undertaken. Statistically speaking, the conceptual framework describes the relationship between specific variables identified in the study. It also outlines the input, process and output of the whole investigation. The conceptual framework is also called the research paradigm. Examples of the Theoretical and the Conceptual Framework The difference between theoretical framework and conceptual framework can be further clarified by the following examples on both concepts: Theoretical Framework: Stimulus elicits response. Conceptual Framework: New teaching method improves students' academic performance. Notice in the illustrative example that the theoretical framework basically differs from the conceptual framework in terms of scope. The theoretical framework describes a broader relationship between things. When stimulus is applied, response is expected. The conceptual framework is much more specific in defining this relationship. The conceptual framework specifies the variables that will have to be explored in the investigation. In this example, the variable " teaching method " represents stimulus while

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• J Grad Med Educ
• v.8(3); 2016 Jul

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

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

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

The Literature Review Defined

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

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

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

Purpose and Importance of the Literature Review

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

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

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

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

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

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

Approaching the Literature Review

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

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

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

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

Locating and Organizing the Literature

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

Human Resources

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

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

Strategies for Finding Related Researcher Publications in Databases and Search Engines

Search Tools and Related Literature

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

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

Box Information Resources

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

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

Getting Organized

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

Citation Managers

Knowing When to Say When

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

Putting It All Together

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

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

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

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

• University of Texas Libraries

Literature Reviews

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

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

Hannah Chapman Tripp : Biology, Neuroscience

Carolyn Cunningham : Human Development & Family Sciences, Psychology, Sociology

Larayne Dallas : Engineering

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
• Last Updated: Oct 26, 2022 2:49 PM
• URL: https://guides.lib.utexas.edu/literaturereviews

• Case Report
• Open access
• Published: 14 May 2024

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

144 Accesses

Metrics details

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.

Peer Review reports

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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