literature review on study methods

State-of-the-art literature review methodology: A six-step approach for knowledge synthesis

• Original Article
• Open access
• Published: 05 September 2022
• Volume 11 , pages 281–288, ( 2022 )

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• Erin S. Barry   ORCID: orcid.org/0000-0003-0788-7153 1 , 2 ,
• Jerusalem Merkebu   ORCID: orcid.org/0000-0003-3707-8920 3 &
• Lara Varpio   ORCID: orcid.org/0000-0002-1412-4341 3  

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Introduction

Researchers and practitioners rely on literature reviews to synthesize large bodies of knowledge. Many types of literature reviews have been developed, each targeting a specific purpose. However, these syntheses are hampered if the review type’s paradigmatic roots, methods, and markers of rigor are only vaguely understood. One literature review type whose methodology has yet to be elucidated is the state-of-the-art (SotA) review. If medical educators are to harness SotA reviews to generate knowledge syntheses, we must understand and articulate the paradigmatic roots of, and methods for, conducting SotA reviews.

We reviewed 940 articles published between 2014–2021 labeled as SotA reviews. We (a) identified all SotA methods-related resources, (b) examined the foundational principles and techniques underpinning the reviews, and (c) combined our findings to inductively analyze and articulate the philosophical foundations, process steps, and markers of rigor.

In the 940 articles reviewed, nearly all manuscripts (98%) lacked citations for how to conduct a SotA review. The term “state of the art” was used in 4 different ways. Analysis revealed that SotA articles are grounded in relativism and subjectivism.

This article provides a 6-step approach for conducting SotA reviews. SotA reviews offer an interpretive synthesis that describes: This is where we are now. This is how we got here. This is where we could be going. This chronologically rooted narrative synthesis provides a methodology for reviewing large bodies of literature to explore why and how our current knowledge has developed and to offer new research directions.

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Literature reviews play a foundational role in scientific research; they support knowledge advancement by collecting, describing, analyzing, and integrating large bodies of information and data [ 1 , 2 ]. Indeed, as Snyder [ 3 ] argues, all scientific disciplines require literature reviews grounded in a methodology that is accurate and clearly reported. Many types of literature reviews have been developed, each with a unique purpose, distinct methods, and distinguishing characteristics of quality and rigor [ 4 , 5 ].

Each review type offers valuable insights if rigorously conducted [ 3 , 6 ]. Problematically, this is not consistently the case, and the consequences can be dire. Medical education’s policy makers and institutional leaders rely on knowledge syntheses to inform decision making [ 7 ]. Medical education curricula are shaped by these syntheses. Our accreditation standards are informed by these integrations. Our patient care is guided by these knowledge consolidations [ 8 ]. Clearly, it is important for knowledge syntheses to be held to the highest standards of rigor. And yet, that standard is not always maintained. Sometimes scholars fail to meet the review’s specified standards of rigor; other times the markers of rigor have never been explicitly articulated. While we can do little about the former, we can address the latter. One popular literature review type whose methodology has yet to be fully described, vetted, and justified is the state-of-the-art (SotA) review.

While many types of literature reviews amalgamate bodies of literature, SotA reviews offer something unique. By looking across the historical development of a body of knowledge, SotA reviews delves into questions like: Why did our knowledge evolve in this way? What other directions might our investigations have taken? What turning points in our thinking should we revisit to gain new insights? A SotA review—a form of narrative knowledge synthesis [ 5 , 9 ]—acknowledges that history reflects a series of decisions and then asks what different decisions might have been made.

SotA reviews are frequently used in many fields including the biomedical sciences [ 10 , 11 ], medicine [ 12 , 13 , 14 ], and engineering [ 15 , 16 ]. However, SotA reviews are rarely seen in medical education; indeed, a bibliometrics analysis of literature reviews published in 14 core medical education journals between 1999 and 2019 reported only 5 SotA reviews out of the 963 knowledge syntheses identified [ 17 ]. This is not to say that SotA reviews are absent; we suggest that they are often unlabeled. For instance, Schuwirth and van der Vleuten’s article “A history of assessment in medical education” [ 14 ] offers a temporally organized overview of the field’s evolving thinking about assessment. Similarly, McGaghie et al. published a chronologically structured review of simulation-based medical education research that “reviews and critically evaluates historical and contemporary research on simulation-based medical education” [ 18 , p. 50]. SotA reviews certainly have a place in medical education, even if that place is not explicitly signaled.

This lack of labeling is problematic since it conceals the purpose of, and work involved in, the SotA review synthesis. In a SotA review, the author(s) collects and analyzes the historical development of a field’s knowledge about a phenomenon, deconstructs how that understanding evolved, questions why it unfolded in specific ways, and posits new directions for research. Senior medical education scholars use SotA reviews to share their insights based on decades of work on a topic [ 14 , 18 ]; their junior counterparts use them to critique that history and propose new directions [ 19 ]. And yet, SotA reviews are generally not explicitly signaled in medical education. We suggest that at least two factors contribute to this problem. First, it may be that medical education scholars have yet to fully grasp the unique contributions SotA reviews provide. Second, the methodology and methods of SotA reviews are poorly reported making this form of knowledge synthesis appear to lack rigor. Both factors are rooted in the same foundational problem: insufficient clarity about SotA reviews. In this study, we describe SotA review methodology so that medical educators can explicitly use this form of knowledge synthesis to further advance the field.

We developed a four-step research design to meet this goal, illustrated in Fig.  1 .

Four-step research design process used for developing a State-of-the-Art literature review methodology

Step 1: Collect SotA articles

To build our initial corpus of articles reporting SotA reviews, we searched PubMed using the strategy (″state of the art review″[ti] OR ″state of the art review*″) and limiting our search to English articles published between 2014 and 2021. We strategically focused on PubMed, which includes MEDLINE, and is considered the National Library of Medicine’s premier database of biomedical literature and indexes health professions education and practice literature [ 20 ]. We limited our search to 2014–2021 to capture modern use of SotA reviews. Of the 960 articles identified, nine were excluded because they were duplicates, erratum, or corrigendum records; full text copies were unavailable for 11 records. All articles identified ( n  = 940) constituted the corpus for analysis.

Step 2: Compile all methods-related resources

EB, JM, or LV independently reviewed the 940 full-text articles to identify all references to resources that explained, informed, described, or otherwise supported the methods used for conducting the SotA review. Articles that met our criteria were obtained for analysis.

To ensure comprehensive retrieval, we also searched Scopus and Web of Science. Additionally, to find resources not indexed by these academic databases, we searched Google (see Electronic Supplementary Material [ESM] for the search strategies used for each database). EB also reviewed the first 50 items retrieved from each search looking for additional relevant resources. None were identified. Via these strategies, nine articles were identified and added to the collection of methods-related resources for analysis.

Step 3: Extract data for analysis

In Step 3, we extracted three kinds of information from the 940 articles papers identified in Step 1. First, descriptive data on each article were compiled (i.e., year of publication and the academic domain targeted by the journal). Second, each article was examined and excerpts collected about how the term state-of-the-art review was used (i.e., as a label for a methodology in-and-of itself; as an adjective qualifying another type of literature review; as a term included in the paper’s title only; or in some other way). Finally, we extracted excerpts describing: the purposes and/or aims of the SotA review; the methodology informing and methods processes used to carry out the SotA review; outcomes of analyses; and markers of rigor for the SotA review.

Two researchers (EB and JM) coded 69 articles and an interrater reliability of 94.2% was achieved. Any discrepancies were discussed. Given the high interrater reliability, the two authors split the remaining articles and coded independently.

Step 4: Construct the SotA review methodology

The methods-related resources identified in Step 2 and the data extractions from Step 3 were inductively analyzed by LV and EB to identify statements and research processes that revealed the ontology (i.e., the nature of reality that was reflected) and the epistemology (i.e., the nature of knowledge) underpinning the descriptions of the reviews. These authors studied these data to determine if the synthesis adhered to an objectivist or a subjectivist orientation, and to synthesize the purposes realized in these papers.

To confirm these interpretations, LV and EB compared their ontology, epistemology, and purpose determinations against two expectations commonly required of objectivist synthesis methods (e.g., systematic reviews): an exhaustive search strategy and an appraisal of the quality of the research data. These expectations were considered indicators of a realist ontology and objectivist epistemology [ 21 ] (i.e., that a single correct understanding of the topic can be sought through objective data collection {e.g., systematic reviews [ 22 ]}). Conversely, the inverse of these expectations were considered indicators of a relativist ontology and subjectivist epistemology [ 21 ] (i.e., that no single correct understanding of the topic is available; there are multiple valid understandings that can be generated and so a subjective interpretation of the literature is sought {e.g., narrative reviews [ 9 ]}).

Once these interpretations were confirmed, LV and EB reviewed and consolidated the methods steps described in these data. Markers of rigor were then developed that aligned with the ontology, epistemology, and methods of SotA reviews.

Of the 940 articles identified in Step 1, 98% ( n  = 923) lacked citations or other references to resources that explained, informed, or otherwise supported the SotA review process. Of the 17 articles that included supporting information, 16 cited Grant and Booth’s description [ 4 ] consisting of five sentences describing the overall purpose of SotA reviews, three sentences noting perceived strengths, and four sentences articulating perceived weaknesses. This resource provides no guidance on how to conduct a SotA review methodology nor markers of rigor. The one article not referencing Grant and Booth used “an adapted comparative effectiveness research search strategy that was adapted by a health sciences librarian” [ 23 , p. 381]. One website citation was listed in support of this strategy; however, the page was no longer available in summer 2021. We determined that the corpus was uninformed by a cardinal resource or a publicly available methodology description.

In Step 2 we identified nine resources [ 4 , 5 , 24 , 25 , 26 , 27 , 28 ]; none described the methodology and/or processes of carrying out SotA reviews. Nor did they offer explicit descriptions of the ontology or epistemology underpinning SotA reviews. Instead, these resources provided short overview statements (none longer than one paragraph) about the review type [ 4 , 5 , 24 , 25 , 26 , 27 , 28 ]. Thus, we determined that, to date, there are no available methodology papers describing how to conduct a SotA review.

Step 3 revealed that “state of the art” was used in 4 different ways across the 940 articles (see Fig.  2 for the frequency with which each was used). In 71% ( n  = 665 articles), the phrase was used only in the title, abstract, and/or purpose statement of the article; the phrase did not appear elsewhere in the paper and no SotA methodology was discussed. Nine percent ( n  = 84) used the phrase as an adjective to qualify another literature review type and so relied entirely on the methodology of a different knowledge synthesis approach (e.g., “a state of the art systematic review [ 29 ]”). In 5% ( n  = 52) of the articles, the phrase was not used anywhere within the article; instead, “state of the art” was the type of article within a journal. In the remaining 15% ( n  = 139), the phrase denoted a specific methodology (see ESM for all methodology articles). Via Step 4’s inductive analysis, the following foundational principles of SotA reviews were developed: (1) the ontology, (2) epistemology, and (3) purpose of SotA reviews.

Four ways the term “state of the art” is used in the corpus and how frequently each is used

Ontology of SotA reviews: Relativism

SotA reviews rest on four propositions:

The literature addressing a phenomenon offers multiple perspectives on that topic (i.e., different groups of researchers may hold differing opinions and/or interpretations of data about a phenomenon).

The reality of the phenomenon itself cannot be completely perceived or understood (i.e., due to limitations [e.g., the capabilities of current technologies, a research team’s disciplinary orientation] we can only perceive a limited part of the phenomenon).

The reality of the phenomenon is a subjective and inter-subjective construction (i.e., what we understand about a phenomenon is built by individuals and so their individual subjectivities shape that understanding).

The context in which the review was conducted informs the review (e.g., a SotA review of literature about gender identity and sexual function will be synthesized differently by researchers in the domain of gender studies than by scholars working in sex reassignment surgery).

As these propositions suggest, SotA scholars bring their experiences, expectations, research purposes, and social (including academic) orientations to bear on the synthesis work. In other words, a SotA review synthesizes the literature based on a specific orientation to the topic being addressed. For instance, a SotA review written by senior scholars who are experts in the field of medical education may reflect on the turning points that have shaped the way our field has evolved the modern practices of learner assessment, noting how the nature of the problem of assessment has moved: it was first a measurement problem, then a problem that embraced human judgment but needed assessment expertise, and now a whole system problem that is to be addressed from an integrated—not a reductionist—perspective [ 12 ]. However, if other scholars were to examine this same history from a technological orientation, learner assessment could be framed as historically constricted by the media available through which to conduct assessment, pointing to how artificial intelligence is laying the foundation for the next wave of assessment in medical education [ 30 ].

Given these foundational propositions, SotA reviews are steeped in a relativist ontology—i.e., reality is socially and experientially informed and constructed, and so no single objective truth exists. Researchers’ interpretations reflect their conceptualization of the literature—a conceptualization that could change over time and that could conflict with the understandings of others.

Epistemology of SotA reviews: Subjectivism

SotA reviews embrace subjectivism. The knowledge generated through the review is value-dependent, growing out of the subjective interpretations of the researcher(s) who conducted the synthesis. The SotA review generates an interpretation of the data that is informed by the expertise, experiences, and social contexts of the researcher(s). Furthermore, the knowledge developed through SotA reviews is shaped by the historical point in time when the review was conducted. SotA reviews are thus steeped in the perspective that knowledge is shaped by individuals and their community, and is a synthesis that will change over time.

Purpose of SotA reviews

SotA reviews create a subjectively informed summary of modern thinking about a topic. As a chronologically ordered synthesis, SotA reviews describe the history of turning points in researchers’ understanding of a phenomenon to contextualize a description of modern scientific thinking on the topic. The review presents an argument about how the literature could be interpreted; it is not a definitive statement about how the literature should or must be interpreted. A SotA review explores: the pivotal points shaping the historical development of a topic, the factors that informed those changes in understanding, and the ways of thinking about and studying the topic that could inform the generation of further insights. In other words, the purpose of SotA reviews is to create a three-part argument: This is where we are now in our understanding of this topic. This is how we got here. This is where we could go next.

The SotA methodology

Based on study findings and analyses, we constructed a six-stage SotA review methodology. This six-stage approach is summarized and guiding questions are offered in Tab.  1 .

Stage 1: Determine initial research question and field of inquiry

In Stage 1, the researcher(s) creates an initial description of the topic to be summarized and so must determine what field of knowledge (and/or practice) the search will address. Knowledge developed through the SotA review process is shaped by the context informing it; thus, knowing the domain in which the review will be conducted is part of the review’s foundational work.

Stage 2: Determine timeframe

This stage involves determining the period of time that will be defined as SotA for the topic being summarized. The researcher(s) should engage in a broad-scope overview of the literature, reading across the range of literature available to develop insights into the historical development of knowledge on the topic, including the turning points that shape the current ways of thinking about a topic. Understanding the full body of literature is required to decide the dates or events that demarcate the timeframe of now in the first of the SotA’s three-part argument: where we are now . Stage 2 is complete when the researcher(s) can explicitly justify why a specific year or event is the right moment to mark the beginning of state-of-the-art thinking about the topic being summarized.

Stage 3: Finalize research question(s) to reflect timeframe

Based on the insights developed in Stage 2, the researcher(s) will likely need to revise their initial description of the topic to be summarized. The formal research question(s) framing the SotA review are finalized in Stage 3. The revised description of the topic, the research question(s), and the justification for the timeline start year must be reported in the review article. These are markers of rigor and prerequisites for moving to Stage 4.

Stage 4: Develop search strategy to find relevant articles

In Stage 4, the researcher(s) develops a search strategy to identify the literature that will be included in the SotA review. The researcher(s) needs to determine which literature databases contain articles from the domain of interest. Because the review describes how we got here , the review must include literature that predates the state-of-the-art timeframe, determined in Stage 2, to offer this historical perspective.

Developing the search strategy will be an iterative process of testing and revising the search strategy to enable the researcher(s) to capture the breadth of literature required to meet the SotA review purposes. A librarian should be consulted since their expertise can expedite the search processes and ensure that relevant resources are identified. The search strategy must be reported (e.g., in the manuscript itself or in a supplemental file) so that others may replicate the process if they so choose (e.g., to construct a different SotA review [and possible different interpretations] of the same literature). This too is a marker of rigor for SotA reviews: the search strategies informing the identification of literature must be reported.

Stage 5: Analyses

The literature analysis undertaken will reflect the subjective insights of the researcher(s); however, the foundational premises of inductive research should inform the analysis process. Therefore, the researcher(s) should begin by reading the articles in the corpus to become familiar with the literature. This familiarization work includes: noting similarities across articles, observing ways-of-thinking that have shaped current understandings of the topic, remarking on assumptions underpinning changes in understandings, identifying important decision points in the evolution of understanding, and taking notice of gaps and assumptions in current knowledge.

The researcher(s) can then generate premises for the state-of-the-art understanding of the history that gave rise to modern thinking, of the current body of knowledge, and of potential future directions for research. In this stage of the analysis, the researcher(s) should document the articles that support or contradict their premises, noting any collections of authors or schools of thinking that have dominated the literature, searching for marginalized points of view, and studying the factors that contributed to the dominance of particular ways of thinking. The researcher(s) should also observe historical decision points that could be revisited. Theory can be incorporated at this stage to help shape insights and understandings. It should be highlighted that not all corpus articles will be used in the SotA review; instead, the researcher(s) will sample across the corpus to construct a timeline that represents the seminal moments of the historical development of knowledge.

Next, the researcher(s) should verify the thoroughness and strength of their interpretations. To do this, the researcher(s) can select different articles included in the corpus and examine if those articles reflect the premises the researcher(s) set out. The researcher(s) may also seek out contradictory interpretations in the literature to be sure their summary refutes these positions. The goal of this verification work is not to engage in a triangulation process to ensure objectivity; instead, this process helps the researcher(s) ensure the interpretations made in the SotA review represent the articles being synthesized and respond to the interpretations offered by others. This is another marker of rigor for SotA reviews: the authors should engage in and report how they considered and accounted for differing interpretations of the literature, and how they verified the thoroughness of their interpretations.

Stage 6: Reflexivity

Given the relativist subjectivism of a SotA review, it is important that the manuscript offer insights into the subjectivity of the researcher(s). This reflexivity description should articulate how the subjectivity of the researcher(s) informed interpretations of the data. These reflections will also influence the suggested directions offered in the last part of the SotA three-part argument: where we could go next. This is the last marker of rigor for SotA reviews: researcher reflexivity must be considered and reported.

SotA reviews have much to offer our field since they provide information on the historical progression of medical education’s understanding of a topic, the turning points that guided that understanding, and the potential next directions for future research. Those future directions may question the soundness of turning points and prior decisions, and thereby offer new paths of investigation. Since we were unable to find a description of the SotA review methodology, we inductively developed a description of the methodology—including its paradigmatic roots, the processes to be followed, and the markers of rigor—so that scholars can harness the unique affordances of this type of knowledge synthesis.

Given their chronology- and turning point-based orientation, SotA reviews are inherently different from other types of knowledge synthesis. For example, systematic reviews focus on specific research questions that are narrow in scope [ 32 , 33 ]; in contrast, SotA reviews present a broader historical overview of knowledge development and the decisions that gave rise to our modern understandings. Scoping reviews focus on mapping the present state of knowledge about a phenomenon including, for example, the data that are currently available, the nature of that data, and the gaps in knowledge [ 34 , 35 ]; conversely, SotA reviews offer interpretations of the historical progression of knowledge relating to a phenomenon centered on significant shifts that occurred during that history. SotA reviews focus on the turning points in the history of knowledge development to suggest how different decisions could give rise to new insights. Critical reviews draw on literature outside of the domain of focus to see if external literature can offer new ways of thinking about the phenomenon of interest (e.g., drawing on insights from insects’ swarm intelligence to better understand healthcare team adaptation [ 36 ]). SotA reviews focus on one domain’s body of literature to construct a timeline of knowledge development, demarcating where we are now, demonstrating how this understanding came to be via different turning points, and offering new research directions. Certainly, SotA reviews offer a unique kind of knowledge synthesis.

Our six-stage process for conducting these reviews reflects the subjectivist relativism that underpins the methodology. It aligns with the requirements proposed by others [ 24 , 25 , 26 , 27 ], what has been written about SotA reviews [ 4 , 5 ], and the current body of published SotA reviews. In contrast to existing guidance [ 4 , 5 , 20 , 21 , 22 , 23 ], our description offers a detailed reporting of the ontology, epistemology, and methodology processes for conducting the SotA review.

This explicit methodology description is essential since many academic journals list SotA reviews as an accepted type of literature review. For instance, Educational Research Review [ 24 ], the American Academy of Pediatrics [ 25 ], and Thorax all lists SotA reviews as one of the types of knowledge syntheses they accept [ 27 ]. However, while SotA reviews are valued by academia, guidelines or specific methodology descriptions for researchers to follow when conducting this type of knowledge synthesis are conspicuously absent. If academics in general, and medical education more specifically, are to take advantage of the insights that SotA reviews can offer, we need to rigorously engage in this synthesis work; to do that, we need clear descriptions of the methodology underpinning this review. This article offers such a description. We hope that more medical educators will conduct SotA reviews to generate insights that will contribute to further advancing our field’s research and scholarship.

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Acknowledgements

We thank Rhonda Allard for her help with the literature review and compiling all available articles. We also want to thank the PME editors who offered excellent development and refinement suggestions that greatly improved this manuscript.

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Department of Anesthesiology, F. Edward Hébert School of Medicine, Uniformed Services University, Bethesda, MD, USA

Erin S. Barry

School of Health Professions Education (SHE), Maastricht University, Maastricht, The Netherlands

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Correspondence to Erin S. Barry .

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E.S. Barry, J. Merkebu and L. Varpio declare that they have no competing interests.

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Supplementary Information

40037_2022_725_moesm1_esm.docx.

For information regarding the search strategy to develop the corpus and search strategy for confirming capture of any available State of the Art review methodology descriptions. Additionally, a list of the methodology articles found through the search strategy/corpus is included

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Barry, E.S., Merkebu, J. & Varpio, L. State-of-the-art literature review methodology: A six-step approach for knowledge synthesis. Perspect Med Educ 11 , 281–288 (2022). https://doi.org/10.1007/s40037-022-00725-9

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Research Methods

• Getting Started
• Literature Review Research
• Research Design
• Research Design By Discipline
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• Teaching with SAGE Research Methods

Literature Review

• What is a Literature Review?
• What is NOT a Literature Review?
• Purposes of a Literature Review
• Types of Literature Reviews
• Literature Reviews vs. Systematic Reviews
• Systematic vs. Meta-Analysis

Literature Review  is a comprehensive survey of the works published in a particular field of study or line of research, usually over a specific period of time, in the form of an in-depth, critical bibliographic essay or annotated list in which attention is drawn to the most significant works.

Also, we can define a literature review as the collected body of scholarly works related to a topic:

• Summarizes and analyzes previous research relevant to a topic
• Includes scholarly books and articles published in academic journals
• Can be an specific scholarly paper or a section in a research paper

The objective of a Literature Review is to find previous published scholarly works relevant to an specific topic

• Help gather ideas or information
• Keep up to date in current trends and findings
• Help develop new questions

A literature review is important because it:

• Explains the background of research on a topic.
• Demonstrates why a topic is significant to a subject area.
• Helps focus your own research questions or problems
• Discovers relationships between research studies/ideas.
• Suggests unexplored ideas or populations
• Identifies major themes, concepts, and researchers on a topic.
• Tests assumptions; may help counter preconceived ideas and remove unconscious bias.
• Identifies critical gaps, points of disagreement, or potentially flawed methodology or theoretical approaches.
• Indicates potential directions for future research.

All content in this section is from Literature Review Research from Old Dominion University 

Keep in mind the following, a literature review is NOT:

Not an essay 

Not an annotated bibliography  in which you summarize each article that you have reviewed.  A literature review goes beyond basic summarizing to focus on the critical analysis of the reviewed works and their relationship to your research question.

Not a research paper   where you select resources to support one side of an issue versus another.  A lit review should explain and consider all sides of an argument in order to avoid bias, and areas of agreement and disagreement should be highlighted.

A literature review serves several purposes. For example, it

• provides thorough knowledge of previous studies; introduces seminal works.
• helps focus one’s own research topic.
• identifies a conceptual framework for one’s own research questions or problems; indicates potential directions for future research.
• suggests previously unused or underused methodologies, designs, quantitative and qualitative strategies.
• identifies gaps in previous studies; identifies flawed methodologies and/or theoretical approaches; avoids replication of mistakes.
• helps the researcher avoid repetition of earlier research.
• suggests unexplored populations.
• determines whether past studies agree or disagree; identifies controversy in the literature.
• tests assumptions; may help counter preconceived ideas and remove unconscious bias.

As Kennedy (2007) notes*, it is important to think of knowledge in a given field as consisting of three layers. First, there are the primary studies that researchers conduct and publish. Second are the reviews of those studies that summarize and offer new interpretations built from and often extending beyond the original studies. Third, there are the perceptions, conclusions, opinion, and interpretations that are shared informally that become part of the lore of field. In composing a literature review, it is important to note that it is often this third layer of knowledge that is cited as "true" even though it often has only a loose relationship to the primary studies and secondary literature reviews.

Given this, while literature reviews are designed to provide an overview and synthesis of pertinent sources you have explored, there are several approaches to how they can be done, depending upon the type of analysis underpinning your study. Listed below are definitions of types of literature reviews:

Argumentative Review      This form examines literature selectively in order to support or refute an argument, deeply imbedded assumption, or philosophical problem already established in the literature. The purpose is to develop a body of literature that establishes a contrarian viewpoint. Given the value-laden nature of some social science research [e.g., educational reform; immigration control], argumentative approaches to analyzing the literature can be a legitimate and important form of discourse. However, note that they can also introduce problems of bias when they are used to to make summary claims of the sort found in systematic reviews.

Integrative Review      Considered a form of research that reviews, critiques, and synthesizes representative literature on a topic in an integrated way such that new frameworks and perspectives on the topic are generated. The body of literature includes all studies that address related or identical hypotheses. A well-done integrative review meets the same standards as primary research in regard to clarity, rigor, and replication.

Historical Review      Few things rest in isolation from historical precedent. Historical reviews are focused on examining research throughout a period of time, often starting with the first time an issue, concept, theory, phenomena emerged in the literature, then tracing its evolution within the scholarship of a discipline. The purpose is to place research in a historical context to show familiarity with state-of-the-art developments and to identify the likely directions for future research.

Methodological Review      A review does not always focus on what someone said [content], but how they said it [method of analysis]. This approach provides a framework of understanding at different levels (i.e. those of theory, substantive fields, research approaches and data collection and analysis techniques), enables researchers to draw on a wide variety of knowledge ranging from the conceptual level to practical documents for use in fieldwork in the areas of ontological and epistemological consideration, quantitative and qualitative integration, sampling, interviewing, data collection and data analysis, and helps highlight many ethical issues which we should be aware of and consider as we go through our study.

Systematic Review      This form consists of an overview of existing evidence pertinent to a clearly formulated research question, which uses pre-specified and standardized methods to identify and critically appraise relevant research, and to collect, report, and analyse data from the studies that are included in the review. Typically it focuses on a very specific empirical question, often posed in a cause-and-effect form, such as "To what extent does A contribute to B?"

Theoretical Review      The purpose of this form is to concretely examine the corpus of theory that has accumulated in regard to an issue, concept, theory, phenomena. The theoretical literature review help establish what theories already exist, the relationships between them, to what degree the existing theories have been investigated, and to develop new hypotheses to be tested. Often this form is used to help establish a lack of appropriate theories or reveal that current theories are inadequate for explaining new or emerging research problems. The unit of analysis can focus on a theoretical concept or a whole theory or framework.

* Kennedy, Mary M. "Defining a Literature."  Educational Researcher  36 (April 2007): 139-147.

All content in this section is from The Literature Review created by Dr. Robert Larabee USC

Robinson, P. and Lowe, J. (2015),  Literature reviews vs systematic reviews.  Australian and New Zealand Journal of Public Health, 39: 103-103. doi: 10.1111/1753-6405.12393

What's in the name? The difference between a Systematic Review and a Literature Review, and why it matters . By Lynn Kysh from University of Southern California

Systematic review or meta-analysis?

A  systematic review  answers a defined research question by collecting and summarizing all empirical evidence that fits pre-specified eligibility criteria.

A  meta-analysis  is the use of statistical methods to summarize the results of these studies.

Systematic reviews, just like other research articles, can be of varying quality. They are a significant piece of work (the Centre for Reviews and Dissemination at York estimates that a team will take 9-24 months), and to be useful to other researchers and practitioners they should have:

• clearly stated objectives with pre-defined eligibility criteria for studies
• explicit, reproducible methodology
• a systematic search that attempts to identify all studies
• assessment of the validity of the findings of the included studies (e.g. risk of bias)
• systematic presentation, and synthesis, of the characteristics and findings of the included studies

Not all systematic reviews contain meta-analysis. 

Meta-analysis is the use of statistical methods to summarize the results of independent studies. By combining information from all relevant studies, meta-analysis can provide more precise estimates of the effects of health care than those derived from the individual studies included within a review.  More information on meta-analyses can be found in  Cochrane Handbook, Chapter 9 .

A meta-analysis goes beyond critique and integration and conducts secondary statistical analysis on the outcomes of similar studies.  It is a systematic review that uses quantitative methods to synthesize and summarize the results.

An advantage of a meta-analysis is the ability to be completely objective in evaluating research findings.  Not all topics, however, have sufficient research evidence to allow a meta-analysis to be conducted.  In that case, an integrative review is an appropriate strategy. 

Some of the content in this section is from Systematic reviews and meta-analyses: step by step guide created by Kate McAllister.

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

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

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

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

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

What are the parts of a lit review?

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

Introduction:

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

Conclusion:

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

How should I organize my lit review?

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

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

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

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

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

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

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

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

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A literature review is a discussion of the literature (aka. the "research" or "scholarship") surrounding a certain topic. A good literature review doesn't simply summarize the existing material, but provides thoughtful synthesis and analysis. The purpose of a literature review is to orient your own work within an existing body of knowledge. A literature review may be written as a standalone piece or be included in a larger body of work.

You can read more about literature reviews, what they entail, and how to write one, using the resources below. 

Am I the only one struggling to write a literature review?

Dr. Zina O'Leary explains the misconceptions and struggles students often have with writing a literature review. She also provides step-by-step guidance on writing a persuasive literature review.

An Introduction to Literature Reviews

Dr. Eric Jensen, Professor of Sociology at the University of Warwick, and Dr. Charles Laurie, Director of Research at Verisk Maplecroft, explain how to write a literature review, and why researchers need to do so. Literature reviews can be stand-alone research or part of a larger project. They communicate the state of academic knowledge on a given topic, specifically detailing what is still unknown.

This is the first video in a whole series about literature reviews. You can find the rest of the series in our SAGE database, Research Methods:

Videos covering research methods and statistics

Identify Themes and Gaps in Literature (with real examples) | Scribbr

Finding connections between sources is key to organizing the arguments and structure of a good literature review. In this video, you'll learn how to identify themes, debates, and gaps between sources, using examples from real papers.

4 Tips for Writing a Literature Review's Intro, Body, and Conclusion | Scribbr

While each review will be unique in its structure--based on both the existing body of both literature and the overall goals of your own paper, dissertation, or research--this video from Scribbr does a good job simplifying the goals of writing a literature review for those who are new to the process. In this video, you’ll learn what to include in each section, as well as 4 tips for the main body illustrated with an example.

• Literature Review This chapter in SAGE's Encyclopedia of Research Design describes the types of literature reviews and scientific standards for conducting literature reviews.
• UNC Writing Center: Literature Reviews This handout from the Writing Center at UNC will explain what literature reviews are and offer insights into the form and construction of literature reviews in the humanities, social sciences, and sciences.
• Purdue OWL: Writing a Literature Review The overview of literature reviews comes from Purdue's Online Writing Lab. It explains the basic why, what, and how of writing a literature review.

Organizational Tools for Literature Reviews

One of the most daunting aspects of writing a literature review is organizing your research. There are a variety of strategies that you can use to help you in this task. We've highlighted just a few ways writers keep track of all that information! You can use a combination of these tools or come up with your own organizational process. The key is choosing something that works with your own learning style.

Citation Managers

Citation managers are great tools, in general, for organizing research, but can be especially helpful when writing a literature review. You can keep all of your research in one place, take notes, and organize your materials into different folders or categories. Read more about citations managers here:

• Manage Citations & Sources

Concept Mapping

Some writers use concept mapping (sometimes called flow or bubble charts or "mind maps") to help them visualize the ways in which the research they found connects.

There is no right or wrong way to make a concept map. There are a variety of online tools that can help you create a concept map or you can simply put pen to paper. To read more about concept mapping, take a look at the following help guides:

• Using Concept Maps From Williams College's guide, Literature Review: A Self-guided Tutorial

Synthesis Matrix

A synthesis matrix is is a chart you can use to help you organize your research into thematic categories. By organizing your research into a matrix, like the examples below, can help you visualize the ways in which your sources connect. 

• Walden University Writing Center: Literature Review Matrix Find a variety of literature review matrix examples and templates from Walden University.
• Writing A Literature Review and Using a Synthesis Matrix An example synthesis matrix created by NC State University Writing and Speaking Tutorial Service Tutors. If you would like a copy of this synthesis matrix in a different format, like a Word document, please ask a librarian. CC-BY-SA 3.0
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What is a literature review?

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

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

Why is it important?

A literature review is important because it:

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

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1. Choose a topic. Define your research question.

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

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

2. Decide on the scope of your review

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

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

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

Make a list of the databases you will search. 

Where to find databases:

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

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

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

Review the literature

Some questions to help you analyze the research:

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

Tips: 

• Review the abstracts carefully.  
• Keep careful notes so that you may track your thought processes during the research process.
• Create a matrix of the studies for easy analysis, and synthesis, across all of the studies.
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The Research Proposal

83 Components of the Literature Review

Krathwohl (2005) suggests and describes a variety of components to include in a research proposal.  The following sections present these components in a suggested template for you to follow in the preparation of your research proposal.

Introduction

The introduction sets the tone for what follows in your research proposal – treat it as the initial pitch of your idea.  After reading the introduction your reader should:

• Understand what it is you want to do;
• Have a sense of your passion for the topic;
• Be excited about the study´s possible outcomes.

As you begin writing your research proposal it is helpful to think of the introduction as a narrative of what it is you want to do, written in one to three paragraphs.  Within those one to three paragraphs, it is important to briefly answer the following questions:

• What is the central research problem?
• How is the topic of your research proposal related to the problem?
• What methods will you utilize to analyze the research problem?
• Why is it important to undertake this research? What is the significance of your proposed research?  Why are the outcomes of your proposed research important, and to whom or to what are they important?

Note : You may be asked by your instructor to include an abstract with your research proposal.  In such cases, an abstract should provide an overview of what it is you plan to study, your main research question, a brief explanation of your methods to answer the research question, and your expected findings. All of this information must be carefully crafted in 150 to 250 words.  A word of advice is to save the writing of your abstract until the very end of your research proposal preparation.  If you are asked to provide an abstract, you should include 5-7 key words that are of most relevance to your study. List these in order of relevance.

Background and significance

The purpose of this section is to explain the context of your proposal and to describe, in detail, why it is important to undertake this research. Assume that the person or people who will read your research proposal know nothing or very little about the research problem.  While you do not need to include all knowledge you have learned about your topic in this section, it is important to ensure that you include the most relevant material that will help to explain the goals of your research.

While there are no hard and fast rules, you should attempt to address some or all of the following key points:

• State the research problem and provide a more thorough explanation about the purpose of the study than what you stated in the introduction.
• Present the rationale for the proposed research study. Clearly indicate why this research is worth doing.  Answer the “so what?” question.
• Describe the major issues or problems to be addressed by your research. Do not forget to explain how and in what ways your proposed research builds upon previous related research.
• Explain how you plan to go about conducting your research.
• Clearly identify the key or most relevant sources of research you intend to use and explain how they will contribute to your analysis of the topic.
• Set the boundaries of your proposed research, in order to provide a clear focus. Where appropriate, state not only what you will study, but what will be excluded from your study.
• Provide clear definitions of key concepts and terms. As key concepts and terms often have numerous definitions, make sure you state which definition you will be utilizing in your research.

Literature Review

This is the most time-consuming aspect in the preparation of your research proposal and it is a key component of the research proposal. As described in Chapter 5 , the literature review provides the background to your study and demonstrates the significance of the proposed research. Specifically, it is a review and synthesis of prior research that is related to the problem you are setting forth to investigate.  Essentially, your goal in the literature review is to place your research study within the larger whole of what has been studied in the past, while demonstrating to your reader that your work is original, innovative, and adds to the larger whole.

As the literature review is information dense, it is essential that this section be intelligently structured to enable your reader to grasp the key arguments underpinning your study. However, this can be easier to state and harder to do, simply due to the fact there is usually a plethora of related research to sift through. Consequently, a good strategy for writing the literature review is to break the literature into conceptual categories or themes, rather than attempting to describe various groups of literature you reviewed.  Chapter V, “ The Literature Review ,” describes a variety of methods to help you organize the themes.

Here are some suggestions on how to approach the writing of your literature review:

• Think about what questions other researchers have asked, what methods they used, what they found, and what they recommended based upon their findings.
• Do not be afraid to challenge previous related research findings and/or conclusions.
• Assess what you believe to be missing from previous research and explain how your research fills in this gap and/or extends previous research

It is important to note that a significant challenge related to undertaking a literature review is knowing when to stop.  As such, it is important to know how to know when you have uncovered the key conceptual categories underlying your research topic.  Generally, when you start to see repetition in the conclusions or recommendations, you can have confidence that you have covered all of the significant conceptual categories in your literature review.  However, it is also important to acknowledge that researchers often find themselves returning to the literature as they collect and analyze their data.  For example, an unexpected finding may develop as one collects and/or analyzes the data and it is important to take the time to step back and review the literature again, to ensure that no other researchers have found a similar finding.  This may include looking to research outside your field.

This situation occurred with one of the authors of this textbook´s research related to community resilience.  During the interviews, the researchers heard many participants discuss individual resilience factors and how they believed these individual factors helped make the community more resilient, overall.  Sheppard and Williams (2016) had not discovered these individual factors in their original literature review on community and environmental resilience. However, when they returned to the literature to search for individual resilience factors, they discovered a small body of literature in the child and youth psychology field. Consequently, Sheppard and Williams had to go back and add a new section to their literature review on individual resilience factors. Interestingly, their research appeared to be the first research to link individual resilience factors with community resilience factors.

Research design and methods

The objective of this section of the research proposal is to convince the reader that your overall research design and methods of analysis will enable you to solve the research problem you have identified and also enable you to accurately and effectively interpret the results of your research. Consequently, it is critical that the research design and methods section is well-written, clear, and logically organized.  This demonstrates to your reader that you know what you are going to do and how you are going to do it.  Overall, you want to leave your reader feeling confident that you have what it takes to get this research study completed in a timely fashion.

Essentially, this section of the research proposal should be clearly tied to the specific objectives of your study; however, it is also important to draw upon and include examples from the literature review that relate to your design and intended methods.  In other words, you must clearly demonstrate how your study utilizes and builds upon past studies, as it relates to the research design and intended methods.  For example, what methods have been used by other researchers in similar studies?

While it is important to consider the methods that other researchers have employed, it is equally important, if not more so, to consider what methods have not been employed but could be.  Remember, the methods section is not simply a list of tasks to be undertaken. It is also an argument as to why and how the tasks you have outlined will help you investigate the research problem and answer your research question(s).

Tips for writing the research design and methods section:

• Specify the methodological approaches you intend to employ to obtain information and the techniques you will use to analyze the data.
• Specify the research operations you will undertake and he way you will interpret the results of those operations in relation to the research problem.
• Go beyond stating what you hope to achieve through the methods you have chosen. State how you will actually do the methods (i.e. coding interview text, running regression analysis, etc.).
• Anticipate and acknowledge any potential barriers you may encounter when undertaking your research and describe how you will address these barriers.
• Explain where you believe you will find challenges related to data collection, including access to participants and information.

Preliminary suppositions and implications

The purpose of this section is to argue how and in what ways you anticipate that your research will refine, revise, or extend existing knowledge in the area of your study. Depending upon the aims and objectives of your study, you should also discuss how your anticipated findings may impact future research.  For example, is it possible that your research may lead to a new policy, new theoretical understanding, or a new method for analyzing data?  How might your study influence future studies?  What might your study mean for future practitioners working in the field?  Who or what may benefit from your study?  How might your study contribute to social, economic, environmental issues?  While it is important to think about and discuss possibilities such as these, it is equally important to be realistic in stating your anticipated findings.  In other words, you do not want to delve into idle speculation.  Rather, the purpose here is to reflect upon gaps in the current body of literature and to describe how and in what ways you anticipate your research will begin to fill in some or all of those gaps.

The conclusion reiterates the importance and significance of your research proposal and it provides a brief summary of the entire proposed study.  Essentially, this section should only be one or two paragraphs in length. Here is a potential outline for your conclusion:

• Discuss why the study should be done. Specifically discuss how you expect your study will advance existing knowledge and how your study is unique.
• Explain the specific purpose of the study and the research questions that the study will answer.
• Explain why the research design and methods chosen for this study are appropriate, and why other design and methods were not chosen.
• State the potential implications you expect to emerge from your proposed study,
• Provide a sense of how your study fits within the broader scholarship currently in existence related to the research problem.

As with any scholarly research paper, you must cite the sources you used in composing your research proposal.  In a research proposal, this can take two forms: a reference list or a bibliography.  A reference list does what the name suggests, it lists the literature you referenced in the body of your research proposal.  All references in the reference list, must appear in the body of the research proposal.  Remember, it is not acceptable to say “as cited in …”  As a researcher you must always go to the original source and check it for yourself.  Many errors are made in referencing, even by top researchers, and so it is important not to perpetuate an error made by someone else. While this can be time consuming, it is the proper way to undertake a literature review.

In contrast, a bibliography , is a list of everything you used or cited in your research proposal, with additional citations to any key sources relevant to understanding the research problem.  In other words, sources cited in your bibliography may not necessarily appear in the body of your research proposal.  Make sure you check with your instructor to see which of the two you are expected to produce.

Overall, your list of citations should be a testament to the fact that you have done a sufficient level of preliminary research to ensure that your project will complement, but not duplicate, previous research efforts. For social sciences, the reference list or bibliography should be prepared in American Psychological Association (APA) referencing format. Usually, the reference list (or bibliography) is not included in the word count of the research proposal. Again, make sure you check with your instructor to confirm.

An Introduction to Research Methods in Sociology Copyright © 2019 by Valerie A. Sheppard is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License , except where otherwise noted.

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• Volume 33, Issue 5
• Equitable and accessible informed healthcare consent process for people with intellectual disability: a systematic literature review
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• http://orcid.org/0000-0002-8498-7329 Manjekah Dunn 1 , 2 ,
• Iva Strnadová 3 , 4 , 5 ,
• Jackie Leach Scully 4 ,
• Jennifer Hansen 3 ,
• Julie Loblinzk 3 , 5 ,
• Skie Sarfaraz 5 ,
• Chloe Molnar 1 ,
• Elizabeth Emma Palmer 1 , 2
• 1 Faculty of Medicine & Health , University of New South Wales , Sydney , New South Wales , Australia
• 2 The Sydney Children's Hospitals Network , Sydney , New South Wales , Australia
• 3 School of Education , University of New South Wales , Sydney , New South Wales , Australia
• 4 Disability Innovation Institute , University of New South Wales , Sydney , New South Wales , Australia
• 5 Self Advocacy Sydney , Sydney , New South Wales , Australia
• Correspondence to Dr Manjekah Dunn, Paediatrics & Child Health, University of New South Wales Medicine & Health, Sydney, New South Wales, Australia; manjekah.dunn{at}unsw.edu.au

Objective To identify factors acting as barriers or enablers to the process of healthcare consent for people with intellectual disability and to understand how to make this process equitable and accessible.

Data sources Databases: Embase, MEDLINE, PsychINFO, PubMed, SCOPUS, Web of Science and CINAHL. Additional articles were obtained from an ancestral search and hand-searching three journals.

Eligibility criteria Peer-reviewed original research about the consent process for healthcare interventions, published after 1990, involving adult participants with intellectual disability.

Synthesis of results Inductive thematic analysis was used to identify factors affecting informed consent. The findings were reviewed by co-researchers with intellectual disability to ensure they reflected lived experiences, and an easy read summary was created.

Results Twenty-three studies were included (1999 to 2020), with a mix of qualitative (n=14), quantitative (n=6) and mixed-methods (n=3) studies. Participant numbers ranged from 9 to 604 people (median 21) and included people with intellectual disability, health professionals, carers and support people, and others working with people with intellectual disability. Six themes were identified: (1) health professionals’ attitudes and lack of education, (2) inadequate accessible health information, (3) involvement of support people, (4) systemic constraints, (5) person-centred informed consent and (6) effective communication between health professionals and patients. Themes were barriers (themes 1, 2 and 4), enablers (themes 5 and 6) or both (theme 3).

Conclusions Multiple reasons contribute to poor consent practices for people with intellectual disability in current health systems. Recommendations include addressing health professionals’ attitudes and lack of education in informed consent with clinician training, the co-production of accessible information resources and further inclusive research into informed consent for people with intellectual disability.

PROSPERO registration CRD42021290548.

• Decision making
• Healthcare quality improvement
• Patient-centred care
• Quality improvement
• Standards of care

Data availability statement

Data are available upon reasonable request. Additional data and materials such as data collection forms, data extraction and analysis templates and QualSyst assessment data can be obtained by contacting the corresponding author.

This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/ .

https://doi.org/10.1136/bmjqs-2023-016113

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What is already known on this topic

People with intellectual disability are frequently excluded from decision-making processes and not provided equal opportunity for informed consent, despite protections outlined in the United Nations Convention on the Rights of Persons with Disabilities.

People with intellectual disability have the capacity and desire to make informed medical decisions, which can improve their well-being, health satisfaction and health outcomes.

What this review study adds

Health professionals lack adequate training in valid informed consent and making reasonable adjustments for people with intellectual disability, and continue to perpetuate assumptions of incapacity.

Health information provided to people with intellectual disability is often inaccessible and insufficient for them to make informed decisions about healthcare.

The role of support people, systemic constraints, a person-centred approach and ineffective healthcare communication also affect informed consent.

How this review might affect research, practice or policy

Health professionals need additional training on how to provide a valid informed consent process for people with intellectual disability, specifically in using accessible health information, making reasonable adjustments (e.g., longer/multiple appointments, options of a support person attending or not, using plain English), involving the individual in discussions, and communicating effectively with them.

Inclusive research is needed to hear the voices and opinions of people with intellectual disability about healthcare decision-making and about informed consent practices in specific healthcare settings.

Introduction

Approximately 1% of the world’s population have intellectual disability. 1 Intellectual disability is medically defined as a group of neurodevelopmental conditions beginning in childhood, with below average cognitive functioning and adaptive behaviour, including limitations in conceptual, social and practical skills. 2 People with intellectual disability prefer an alternative strength-based definition, reflected in the comment by Robert Strike OAM (Order of Australia Medal): ‘We can learn if the way of teaching matches how the person learns’, 3 reinforcing the importance of providing information tailored to the needs of a person with intellectual disability. A diagnosis of intellectual disability is associated with significant disparities in health outcomes. 4–7 Person-centred decision-making and better communication have been shown to improve patient satisfaction, 8 9 the physician–patient relationship 10 and overall health outcomes 11 for the wider population. Ensuring people with intellectual disability experience informed decision-making and accessible healthcare can help address the ongoing health disparities and facilitate equal access to healthcare.

Bodily autonomy is an individual’s power and agency to make decisions about their own body. 12 Informed consent for healthcare enables a person to practice bodily autonomy and is protected, for example, by the National Safety and Quality Health Service Standards (Australia), 13 Mental Capacity Act (UK) 14 and the Joint Commission Standards (USA). 15 In this article, we define informed consent according to three requirements: (1) the person is provided with information they understand, (2) the decision is free of coercion and (3) the person must have capacity. 16 For informed consent to be valid, this process must be suited to the individual’s needs so that they can understand and communicate effectively. Capacity is the ability to give informed consent for a medical intervention, 17 18 and the Mental Capacity Act outlines that ‘a person must be assumed to have capacity unless it is established that he lacks capacity’ and that incapacity can only be established if ‘all practicable steps’ to support capacity have been attempted without success. 14 These assumptions of capacity are also decision-specific, meaning an individual’s ability to consent can change depending on the situation, the choice itself and other factors. 17

Systemic issues with healthcare delivery systems have resulted in access barriers for people with intellectual disability, 19 despite the disability discrimination legislation in many countries who are signatories to the United Nations (UN) Convention on the Rights of Persons with Disabilities. 20 Patients with intellectual disability are not provided the reasonable adjustments that would enable them to give informed consent for medical procedures or interventions, 21 22 despite evidence that many people with intellectual disability have both the capacity and the desire to make their own healthcare decisions. 21 23

To support people with intellectual disability to make independent health decisions, an equitable and accessible informed consent process is needed. 24 However, current health systems have consistently failed to provide this. 21 25 To address this gap, we must first understand the factors that contribute to inequitable and inaccessible consent. To the best of our knowledge, the only current review of informed consent for people with intellectual disability is an integrative review by Goldsmith et al . 26 Many of the included articles focused on assessment of capacity 27–29 and research consent. 30–32 The review’s conclusion supported the functional approach to assess capacity, with minimal focus on how the informed consent processes can be improved. More recently, there has been a move towards ensuring that the consent process is accessible for all individuals, including elderly patients 33 and people with aphasia. 34 However, there remains a paucity of literature about the informed consent process for people with intellectual disability, with no systematic reviews summarising the factors influencing the healthcare consent process for people with intellectual disability.

To identify barriers to and enablers of the informed healthcare consent process for people with intellectual disability, and to understand how this can be made equitable and accessible.

A systematic literature review was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-Analysis Protocols (PRISMA-P) systematic literature review protocol. 35 The PRISMA 2020 checklist 36 and ENhancing Transparency in REporting the synthesis of Qualitative research (ENTREQ) reporting guidelines were also followed. 37 The full study protocol is included in online supplemental appendix 1 .

Supplemental material

No patients or members of the public were involved in this research for this manuscript.

Search strategy

A search strategy was developed to identify articles about intellectual disability, consent and healthcare interventions, described in online supplemental appendix 2 . Multiple databases were searched for articles published between January 1990 to January 2022 (Embase, MEDLINE, PsychINFO, PubMed, SCOPUS, Web of Science and CINAHL). These databases include healthcare and psychology databases that best capture relevant literature on this topic, including medical, nursing, social sciences and bioethical literature. The search was limited to studies published from 1990 as understandings of consent have changed since then. 38 39 This yielded 4853 unique papers which were imported into Covidence, a specialised programme for conducting systematic reviews. 40

Study selection

Citation screening by abstract and titles was completed by two independent researchers (MD and EEP). Included articles had to:

Examine the informed consent process for a healthcare intervention for people with intellectual disability.

Have collected more than 50% of its data from relevant stakeholders, including adults with intellectual disability, families or carers of a person with intellectual disability, and professionals who engage with people with intellectual disability.

Report empirical data from primary research methodology.

Be published in a peer-reviewed journal after January 1990.

Be available in English.

Full text screening was completed by two independent researchers (MD and EEP). Articles were excluded if consent was only briefly discussed or if it focused on consent for research, capacity assessment, or participant knowledge or comprehension. Any conflicts were resolved through discussion with an independent third researcher (IS).

Additional studies were identified through an ancestral search and by hand-searching three major journals relevant to intellectual disability research. Journals were selected if they had published more than one included article for this review or in previous literature reviews conducted by the research team.

Quality assessment

Two independent researchers (MD and IS) assessed study quality with the QualSyst tool, 41 which can assess both qualitative and quantitative research papers. After evaluating the distribution of scores, a threshold value of 55% was used, as suggested by QualSyst 41 to exclude poor-quality studies but capture enough studies overall. Any conflicts between the quality assessment scores were resolved by a third researcher (EEP). For mixed-method studies, both qualitative and quantitative quality scores were calculated, and the higher value used.

Data collection

Two independent researchers (MD and JH) reviewed each study and extracted relevant details, including study size, participant demographics, year, country of publication, study design, data analysis and major outcomes reported. Researchers used standardised data collection forms designed, with input from senior researchers with expertise in qualitative research (IS and EEP), to extract data relevant to the review’s research aims. The form was piloted on one study, and a second iteration made based on feedback. These forms captured data on study design, methods, participants, any factors affecting the process of informed consent and study limitations. Data included descriptions and paragraphs outlining key findings, the healthcare context, verbatim participant quotes and any quantitative analyses or statistics. Missing or unclear data were noted.

Data analysis

A pilot literature search showed significant heterogeneity in methodology of studies, limiting the applicability of traditional quantitative analysis (ie, meta-analysis). Instead, inductive thematic analysis was chosen as an alternative methodology 42 43 that has been used in recent systematic reviews examining barriers and enablers of other health processes. 44 45 The six-phase approach described by Braun and Clarke was used. 46 47 A researcher (MD) independently coded the extracted data of each study line-by-line, with subsequent data grouped into pre-existing codes or new concepts when necessary. Codes were reviewed iteratively and grouped into categories, subthemes and themes framed around the research question. Another independent researcher (JH) collated and analysed the data on study demographics, methods and limitations. The themes were reviewed by two senior researchers (EEP and IS).

Qualitative methods of effect size calculations have been described in the literature, 48 49 which was captured in this review by the number of studies that identified each subtheme, with an assigned frequency rating to compare their relative significance. Subthemes were given a frequency rating of A, B, C or D if they were identified by >10, 7–9, 4–6 or <3 articles, respectively. The overall significance of each theme was estimated by the number of studies that mentioned it and the GRADE framework, a stepwise approach to quality assessment using a four-tier rating system. Each study was evaluated for risk of bias, inconsistency, indirectness, imprecision and publication bias. 50 51 Study sensitivity was assessed by counting the number of distinct subthemes included. 52 The quality of findings was designated high, moderate or low depending on the frequency ratings, the QualSyst score and the GRADE scores of studies supporting the finding. Finally, the relative contributions of each study were evaluated by the number of subthemes described, guided by previously reported methods for qualitative reviews. 52

Co-research

The findings were reviewed by two co-researchers with intellectual disability (JL and SS), with over 30 years combined experience as members and employees of a self-advocacy organisation. Guidance on the findings and an easy read summary was produced in line with best-practice inclusive research 53 54 over multiple discussions. Input from two health professional researchers (MD and EEP) provided data triangulation and sense-checking of findings.

Twenty-three articles were identified ( figure 1 ): 14 qualitative, 6 quantitative and 3 mixed-methods. Two papers included the same population of study participants: McCarthy 55 and McCarthy, 56 but had different research questions. Fovargue et al 57 was excluded due to a quality score of 35%. Common quality limitations were a lack of verification procedures to establish credibility and limited researcher reflexivity. No studies were excluded due to language requirements (as all were in English) or age restrictions (all studies had majority adult participants).

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PRISMA 2020 flowchart for the systematic review. 36

Studies were published from 1999 to 2020 and involved participant populations from the UK (n=18), USA (n=3), Sweden (n=1) and Ireland (n=1). Participant numbers ranged from 9 to 604 (median 21), and participants included people with intellectual disability (n=817), health professionals (n=272), carers and support people (n=48), and other professionals that work with people with intellectual disability (n=137, community service agency directors, social workers, administrative staff and care home staff). Ages of participants ranged from 8 to 84 years, though only Aman et al 58 included participants <18 years of age. This study was included as the article states very few children were included. Studies examined consent in different contexts, including contraception and sexual health (6/23 articles), 58–60 medications (5/23 articles), 58–62 emergency healthcare, 63 cervical screening, 64 community referrals, 58–61 65 mental health, 66 hydrotherapy, 64 blood collection 67 and broad decision-making consent without a specific context. 65 68–71 A detailed breakdown of each study is included in online supplemental appendix 3 .

Six major themes were identified from the studies, summarised in figure 2 . An overview of included studies showing study sensitivity, effect size, QualSyst and GRADE scores is given in online supplemental appendix 4 . Studies with higher QualSyst and GRADE scores contributed more to this review’s findings and tended to include more subthemes; specifically, Rogers et al , 66 Sowney and Barr, 63 Höglund and Larsson, 72 and McCarthy 55 and McCarthy. 56 Figure 3 gives the easy read version of theme 1, with the full easy read summary in online supplemental appendix 5 .

Summary of the identified six themes and subthemes.

Theme 1 of the easy read summary.

Theme 1—Health professionals’ attitudes and lack of education about informed consent

Health professionals’ attitudes and practices were frequently (18/21) identified as factors affecting the informed consent process, with substantial evidence supporting this theme. Studies noted the lack of training for health professionals in supporting informed consent for people with intellectual disability, their desire for further education, and stereotypes and discrimination perpetuated by health professionals.

Lack of health professional education on informed consent and disability discrimination legislation

Multiple studies reported inconsistent informed consent practices, for various reasons: some reported that health professionals ‘forgot’ to or ‘did not realise consent was necessary’, 63 73 but inconsistent consent practices were also attributed to healthcare providers’ unfamiliarity with consent guidelines and poor education on this topic. Carlson et al 73 reported that only 44% of general practitioners (GPs) were aware of consent guidelines, and there was the misconception that consent was unnecessary for people with intellectual disability. Similarly, studies of psychologists 66 and nurses 63 found that many were unfamiliar with their obligations to obtain consent, despite the existence of anti-discrimination legislation. People with intellectual disability describe feeling discriminated against by health professionals, reflected in comments such as ‘I can tell, my doctor just thinks I’m stupid – I'm nothing to him’. 74 Poor consent practices by health professionals were observed in Goldsmith et al , 67 while health professionals surveyed by McCarthy 56 were unaware of their responsibility to provide accessible health information to women with intellectual disability. Improving health professional education and training was suggested by multiple studies as a way to remove this barrier. 63 65–67 69 73

Lack of training on best practices for health professions caring for people with intellectual disability

A lack of training in caring for and communicating with people with intellectual disability was also described by midwives, 72 psychologists, 66 nurses, 63 pharmacists 61 and GPs. 56 72 75 Health professionals lacked knowledge about best practice approaches to providing equitable healthcare consent processes through reasonable adjustments such as accessible health information, 56 60 66 longer appointments times, 60 72 simple English 62 67 and flexible approaches to patient needs. 63 72

Health professionals’ stereotyping and assumptions of incapacity

Underlying stereotypes contributed to some health professionals’ (including nurses, 63 GPs 56 and physiotherapists 64 ) belief that people with intellectual disability lack capacity and therefore, do not require opportunities for informed consent. 56 64 In a survey of professionals referring people with intellectual disability to a disability service, the second most common reason for not obtaining consent was ‘patient unable to understand’. 73

Proxy consent as an inappropriate alternative

People with intellectual disability are rarely the final decision-maker in their medical choices, with many health providers seeking proxy consent from carers, support workers and family members, despite its legal invalidity. In McCarthy’s study (2010), 18/23 women with intellectual disability said the decision to start contraception was made by someone else. Many GPs appeared unaware that proxy consent is invalid in the UK. 56 Similar reports came from people with intellectual disability, 55 56 60 64 69 76 health professionals (nurses, doctors, allied health, psychologists), 56 63 64 66 77 support people 64 77 and non-medical professionals, 65 73 and capacity was rarely documented. 56 62 77

Exclusion of people with intellectual disability from decision-making discussions

Studies described instances where health professionals made decisions for their patients with intellectual disability or coerced patients into a choice. 55 72 74 76 77 In Ledger et al 77 , only 62% of women with intellectual disability were involved in the discussion about contraception, and only 38% made the final decision, and others stated in Wiseman and Ferrie 74 : ‘I was not given the opportunity to explore the different options. I was told what one I should take’. Three papers outlined instances where the choices of people with intellectual disability were ignored despite possessing capacity 65 66 69 and when a procedure continued despite them withdrawing consent. 69

Theme 2—Inadequate accessible health information

Lack of accessible health information.

The lack of accessible health information was the most frequently identified subtheme (16/23 studies). Some studies reported that health professionals provided information to carers instead, 60 avoided providing easy read information due to concerns about ‘offending’ patients 75 or only provided verbal information. 56 67 Informed consent was supported when health professionals recognised the importance of providing medical information 64 and when it was provided in an accessible format. 60 Alternative approaches to health information were explored, including virtual reality 68 and in-person education sessions, 59 with varying results. Overall, the need to provide information in different formats tailored to an individual’s communication needs, rather than a ‘one size fits all’ approach, was emphasised by both people with intellectual disability 60 and health professionals. 66

Insufficient information provided

Studies described situations where insufficient information was provided to people with intellectual disability to make informed decisions. For example, some people felt the information from their GP was often too basic to be helpful (Fish et al 60 ) and wanted additional information on consent forms (Rose et al 78 ).

Theme 3—The involvement of support people

Support people (including carers, family members and group home staff) were identified in 11 articles as both enablers of and barriers to informed consent. The antagonistic nature of these findings and lower frequency of subthemes are reflected in the lower quality assessments of evidence.

Support people facilitated communication with health professionals

Some studies reported carers bridging communication barriers with health to support informed consent. 63 64 McCarthy 56 found 21/23 of women with intellectual disability preferred to see doctors with a support person due to perceived benefits: ‘Sometimes I don’t understand it, so they have to explain it to my carer, so they can explain it to me easier’. Most GPs in this study (93%) also agreed that support people aided communication.

Support people helped people with intellectual disability make decisions

By advocating for people with intellectual disability, carers encouraged decision-making, 64 74 provided health information, 74 77 emotional support 76 and assisted with reading or remembering health information. 55 58 76 Some people with intellectual disability explicitly appreciated their support person’s involvement, 60 such as in McCarthy’s 55 study where 18/23 participants felt supported and safer when a support person was involved.

Support people impeded individual autonomy

The study by Wiseman and Ferrie 74 found that while younger participants with intellectual disability felt family members empowered their decision-making, older women felt family members impaired their ability to give informed consent. This was reflected in interviews with carers who questioned the capacity of the person with intellectual disability they supported and stated they would guide them to pick the ‘best choice’ or even over-ride their choices. 64 Studies of psychologists and community service directors described instances where the decision of family or carers was prioritised over the wishes of the person with intellectual disability. 65 66 Some women with intellectual disability in McCarthy’s studies (2010, 2009) 55 56 appeared to have been coerced into using contraception by parental pressures or fear of losing group home support.

Theme 4—Systemic constraints within healthcare systems

Time restraints affect informed consent and accessible healthcare.

Resource limitations create time constraints that impair the consent process and have been identified as a barrier by psychologists, 66 GPs, 56 hospital nurses 63 and community disability workers. 73 Rogers et al 66 highlighted that a personalised approach that could improve informed decision-making is restricted by inflexible medical models. Only two studies described flexible patient-centred approaches to consent. 60 72 A survey of primary care practices in 2007 reported that most did not modify their cervical screening information for patients with intellectual disability because it was not practical. 75

Inflexible models of consent

Both people with intellectual disability 76 and health professionals 66 recognised that consent is traditionally obtained through one-off interactions prior to an intervention. Yet, for people with intellectual disability, consent should ideally be an ongoing process that begins before an appointment and continues between subsequent ones. Other studies have tended to describe one-off interactions where decision-making was not revisited at subsequent appointments. 56 60 72 76

Lack of systemic supports

In one survey, self-advocates highlighted a lack of information on medication for people with intellectual disability and suggested a telephone helpline and a centralised source of information to support consent. 60 Health professionals also want greater systemic support, such as a health professional specialised in intellectual disability care to support other staff, 72 or a pharmacist specifically to help patients with intellectual disability. 61 Studies highlighted a lack of guidelines about healthcare needs of people with intellectual disabilities such as contraceptive counselling 72 or primary care. 75

Theme 5—Person-centred informed consent

Ten studies identified factors related to a person-centred approach to informed consent, grouped below into three subthemes. Health professionals should tailor their practice when obtaining informed consent from people with intellectual disability by considering how these subthemes relate to the individual. Each subtheme was described five times in the literature with a relative frequency rating of ‘C’, contributing to overall lower quality scores.

Previous experience with decision-making

Arscott et al 71 found that the ability of people with intellectual disability to consent changed with their verbal and memory skills and in different clinical vignettes, supporting the view of ‘functional’ capacity specific to the context of the medical decision. Although previous experiences with decision-making did not influence informed consent in this paper, other studies suggest that people with intellectual disability accustomed to independent decision-making were more able to make informed medical decisions, 66 70 and those who live independently were more likely to make independent healthcare decisions. 56 Health professionals should be aware that their patients with intellectual disability will have variable experience with decision-making and provide individualised support to meet their needs.

Variable awareness about healthcare rights

Consent processes should be tailored to the health literacy of patients, including emphasising available choices and the option to refuse treatment. In some studies, medical decisions were not presented to people with intellectual disability as a choice, 64 and people with intellectual disability were not informed of their legal right to accessible health information. 56

Power differences and acquiescence

Acquiescence by people with intellectual disability due to common and repeated experiences of trauma—that is, their tendency to agree with suggestions made by carers and health professionals, often to avoid upsetting others—was identified as an ongoing barrier. In McCarthy’s (2009) interviews with women with intellectual disability, some participants implicitly rejected the idea that they might make their own healthcare decisions: ‘They’re the carers, they have responsibility for me’. Others appeared to have made decisions to appease their carers: ‘I have the jab (contraceptive injection) so I can’t be blamed for getting pregnant’. 55 Two studies highlighted that health professionals need to be mindful of power imbalances when discussing consent with people with intellectual disability to ensure the choices are truly autonomous. 61 66

Theme 6—Effective communication between health professionals and patients

Implementation of reasonable adjustments for verbal and written information.

Simple language was always preferred by people with intellectual disability. 60 67 Other communication aids used in decision-making included repetition, short sentences, models, pictures and easy read brochures. 72 Another reasonable adjustment is providing the opportunity to ask questions, which women with intellectual disability in McCarthy’s (2009) study reported did not occur. 55

Tailored communication methods including non-verbal communication

Midwives noted that continuity of care allows them to develop rapport and understand the communication preferences of people with intellectual disability. 72 This is not always possible; for emergency nurses, the lack of background information about patients with intellectual disability made it challenging to understand their communication preferences. 63 The use of non-verbal communication, such as body language, was noted as underutilised 62 66 and people with intellectual disability supported the use of hearing loops, braille and sign language. 60

To the best of our knowledge, this is the first systematic review investigating the barriers and enablers of the informed consent process for healthcare procedures for people with intellectual disability. The integrative review by Goldsmith et al 26 examined capacity assessment and shares only three articles with this systematic review. 69 71 73 Since the 2000s, there has been a paradigm shift in which capacity is no longer considered a fixed ability that only some individuals possess 38 39 but instead as ‘functional’: a flexible ability that changes over time and in different contexts, 79 reflected in Goldsmith’s review. An individual’s capacity can be supported through various measures, including how information is communicated and how the decision-making process is approached. 18 80 By recognising the barriers and enablers identified in this review, physicians can help ensure the consent process for their patients with intellectual disability is both valid and truly informed. This review has highlighted the problems of inaccessible health information, insufficient clinical education on how to make reasonable adjustments and lack of person-centred trauma-informed care.

Recommendations

Health professionals require training in the informed consent process for people with intellectual disability, particularly in effective and respectful communication, reasonable adjustments and trauma-informed care. Reasonable adjustments include offering longer or multiple appointments, using accessible resources (such as easy read information or shared decision-making tools) and allowing patient choices (such as to record a consultation or involve a support person). Co-researchers reported that many people with intellectual disability prefer to go without a support person because they find it difficult to challenge their decisions and feel ignored if the health professional only talks to the support person. People with intellectual disability also feel they cannot seek second opinions before making medical decisions or feel pressured to provide consent, raising the possibility of coercion. These experiences contribute to healthcare trauma. Co-researchers raised the importance of building rapport with the person with intellectual disability and of making reasonable adjustments, such as actively advocating for the person’s autonomy, clearly stating all options including the choice to refuse treatment, providing opportunities to contribute to discussions and multiple appointments to ask questions and understand information. They felt that without these efforts to support consent, health professionals can reinforce traumatic healthcare experiences for people with intellectual disability. Co-researchers noted instances where choices were made by doctors without discussion and where they were only given a choice after requesting one and expressed concern that these barriers are greater for those with higher support needs.

Co-researchers showed how these experiences contributed to mistrust of health professionals and poorer health outcomes. In one situation, a co-researcher was not informed of a medication’s withdrawal effects, resulting in significant side-effects when it was ceased. Many people with intellectual disability describe a poor relationship with their health professionals, finding it difficult to trust health information provided due to previous traumatic experiences of disrespect, coercion, lack of choice and inadequate support. Many feel they cannot speak up due to the power imbalance and fear of retaliation. Poor consent practices and lack of reasonable adjustments directly harm therapeutic alliances by reducing trust, contribute to healthcare trauma and lead to poorer health outcomes for people with intellectual disability.

Additional education and training for health professionals is urgently needed in the areas of informed consent, reasonable adjustments and effective communication with people with intellectual disability. The experiences of health professionals within the research team confirmed that there is limited training in providing high-quality healthcare for people with intellectual disability, including reasonable adjustments and accessible health information. Co-researchers also suggested that education should be provided to carers and support people to help them better advocate for people with intellectual disability.

Health information should be provided in a multimodal format, including written easy read information. Many countries have regulation protecting the right to accessible health information and communication support to make an informed choice, such as UK’s Accessible Information Standard, 81 and Australia’s Charter of Health Care Rights, 24 yet these are rarely observed. Steps to facilitate this include routinely asking patients about information requirements, system alerts for an individual’s needs or routinely providing reasonable adjustments. 82 Co-researchers agreed that there is a lack of accessible health information, particularly about medications, and that diagrams and illustrations are underutilised. There is a critical need for more inclusive and accessible resources to help health professionals support informed consent in a safe and high-quality health system. These resources should be created through methods of inclusive research, such as co-production, actively involving people with intellectual disability in the planning, creation, and feedback process. 53

Strengths and limitations

This systematic review involved two co-researchers with intellectual disability in sense-checking findings and co-creating the easy read summary. Two co-authors who are health professionals provided additional sense-checking of findings from a different stakeholder perspective. In future research, this could be extended by involving people with intellectual disability in the design and planning of the study as per recommendations for best-practice inclusive research. 53 83

The current literature is limited by low use of inclusive research practices in research involving people with intellectual disability, increasing vulnerability to external biases (eg, inaccessible questionnaires, involvement of carers in data collection, overcompliance or acquiescence and absence of researcher reflexivity). Advisory groups or co-research with people with intellectual disability were only used in five studies. 58 60 68 74 76 Other limitations include unclear selection criteria, low sample sizes, missing data, using gatekeepers in patient selection and predominance of UK-based studies—increasing the risk of bias and reducing transferability. Nine studies (out of 15 involving people with intellectual disability) explicitly excluded those with severe or profound intellectual disability, reflecting a selection bias; only one study specifically focused on people with intellectual disability with higher support needs. Studies were limited to a few healthcare contexts, with a focus on consent about sexual health, contraception and medications.

The heterogeneity and qualitative nature of studies made it challenging to apply traditional meta-analysis. However, to promote consistency in qualitative research, the PRISMA and ENTREQ guidelines were followed. 36 37 Although no meta-analyses occurred, the duplication of study populations in McCarthy 2009 and 2010 likely contributed to increased significance of findings reported in both studies. Most included studies (13/23) were published over 10 years ago, reducing the current relevance of this review’s findings. Nonetheless, the major findings reflect underlying systemic issues within the health system, which are unlikely to have been resolved since the articles were published, as the just-released final report of the Australian Royal Commission into Violence, Abuse, Neglect and Exploitation of People with Disability highlights. 84 There is an urgent need for more inclusive studies to explore the recommendations and preferences of people with intellectual disability about healthcare choices.

Informed consent processes for people with intellectual disability should include accessible information and reasonable adjustments, be tailored to individuals’ needs and comply with consent and disability legislation. Resources, guidelines and healthcare education are needed and should cover how to involve carers and support people, address systemic healthcare problems, promote a person-centred approach and ensure effective communication. These resources and future research must use principles of inclusive co-production—involving people with intellectual disability at all stages. Additionally, research is needed on people with higher support needs and in specific contexts where informed consent is vital but under-researched, such as cancer screening, palliative care, prenatal and newborn screening, surgical procedures, genetic medicine and advanced therapeutics such as gene-based therapies.

Ethics statements

Patient consent for publication.

Not applicable.

Ethics approval

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Supplementary materials

Supplementary data.

This web only file has been produced by the BMJ Publishing Group from an electronic file supplied by the author(s) and has not been edited for content.

• Data supplement 1
• Data supplement 2
• Data supplement 3
• Data supplement 4
• Data supplement 5

Contributors MD, EEP and IS conceived the idea for the systematic review. MD drafted the search strategy which was refined by EEP and IS. MD and EEP completed article screening. MD and IS completed quality assessments of included articles. MD and JH completed data extraction. MD drafted the original manuscript. JL and SS were co-researchers who sense-checked findings and were consulted to formulate dissemination plans. JL and SS co-produced the easy read summary with MD, CM, JH, EEP and IS. MD, JLS, EEP and IS reviewed manuscript wording. All authors critically reviewed the manuscript and approved it for publication. The corresponding author attests that all listed authors meet authorship criteria and that no others meeting the criteria have been omitted. MD is the guarantor responsible for the overall content of this manuscript.

Funding This systematic literature review was funded by the National Health & Medical Research Council (NHMRC), Targeted Call for Research (TCR) into Improving health of people with intellectual disability. Research grant title "GeneEQUAL: equitable and accessible genomic healthcare for people with intellectual disability". NHMRC application ID: 2022/GNT2015753.

Competing interests None declared.

Provenance and peer review Not commissioned; externally peer reviewed.

Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.

Linked Articles

• Editorial It is up to healthcare professionals to talk to us in a way that we can understand: informed consent processes in people with an intellectual disability Jonathon Ding Richard Keagan-Bull Irene Tuffrey-Wijne BMJ Quality & Safety 2024; 33 277-279 Published Online First: 30 Jan 2024. doi: 10.1136/bmjqs-2023-016830

Read the full text or download the PDF:

• Study Protocol
• Open access
• Published: 30 April 2024

Co-design and mixed methods evaluation of an interdisciplinary digital resource for undergraduate health profession students to improve the prevention, recognition, and management of delirium in Ireland: a study protocol

• Lana Cook   ORCID: orcid.org/0009-0009-0905-4850 1 ,
• Alice Coffey   ORCID: orcid.org/0000-0002-5178-6723 2 ,
• Christine Brown Wilson   ORCID: orcid.org/0000-0002-7861-9538 1 ,
• Pauline Boland   ORCID: orcid.org/0000-0003-0218-327X 3 ,
• Patrick Stark   ORCID: orcid.org/0000-0003-2659-0865 1 ,
• Margaret Graham   ORCID: orcid.org/0000-0003-4705-0219 2 ,
• James McMahon   ORCID: orcid.org/0000-0003-1418-6611 1 ,
• Dympna Tuohy   ORCID: orcid.org/0000-0001-6240-0315 2 ,
• Heather E Barry   ORCID: orcid.org/0000-0002-9684-8182 4 ,
• Jill Murphy 2 ,
• Matt Birch 1 ,
• Audrey Tierney 3 ,
• Tara Anderson   ORCID: orcid.org/0009-0005-3611-3431 1 ,
• Arlene McCurtin   ORCID: orcid.org/0000-0002-0649-0448 3 ,
• Emma Cunningham 5 ,
• Geoffrey M. Curran   ORCID: orcid.org/0000-0003-3464-8422 6 &
• Gary Mitchell   ORCID: orcid.org/0000-0003-2133-2998 1  

BMC Medical Education volume  24 , Article number:  475 ( 2024 ) Cite this article

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Delirium is a common symptom of acute illness which is potentially avoidable with early recognition and intervention. Despite being a growing concern globally, delirium remains underdiagnosed and poorly reported, with limited understanding of effective delirium education for undergraduate health profession students. Digital resources could be an effective approach to improving professional knowledge of delirium, but studies utilising these with more than one profession are limited, and no evidence-based, interdisciplinary, digital delirium education resources are reported. This study aims to co-design and evaluate a digital resource for undergraduate health profession students across the island of Ireland to improve their ability to prevent, recognise, and manage delirium alongside interdisciplinary colleagues.

Utilising a logic model, three workstreams have been identified. Workstream 1 will comprise three phases: (1) a systematic review identifying the format, methods, and content of existing digital delirium education interventions for health profession students, and their effect on knowledge, self-efficacy, and behavioural change; (2) focus groups with health profession students to determine awareness and experiences of delirium care; and (3) a Delphi survey informed by findings from the systematic review, focus groups, and input from the research team and expert reference group to identify resource priorities. Workstream 2 will involve the co-design of the digital resource through workshops ( n  = 4) with key stakeholders, including health profession students, professionals, and individuals with lived experience of delirium. Lastly, Workstream 3 will involve a mixed methods evaluation of the digital resource. Outcomes include changes to delirium knowledge and self-efficacy towards delirium care, and health profession students experience of using the resource.

Given the dearth of interdisciplinary educational resources on delirium for health profession students, a co-designed, interprofessional, digital education resource will be well-positioned to shape undergraduate delirium education. This research may enhance delirium education and the self-efficacy of future health professionals in providing delirium care, thereby improving practice and patients’ experiences and outcomes.

Trial registration

Not applicable.

Peer Review reports

Delirium is an acute condition characterised by a rapid onset of cognitive decline with fluctuating symptoms. It can be triggered by a wide range of external stimuli such as surgery, infection, head injury, stroke, medication, substance withdrawal, deranged electrolytes and sleep deprivation [ 1 ]. Other features of delirium include altered consciousness, disorganised thoughts, disorientation, impaired memory, inattention, and paranoid delusions and hallucinations [ 2 , 3 ]. Higher-risk groups include hospitalised older adults, children [ 4 ] and people requiring critical care; up to 80% of people admitted to the intensive care unit (ICU) experience delirium [ 5 ] and 30–80% of older adults experience delirium following major surgery [ 6 ]. Delirium can be profoundly distressing for patients, families and staff [ 1 , 3 ]. Persistent exposure to delirium may indicate a symptom of diminished cognitive reserve, potentially heightening susceptibility to the development of dementia [ 7 , 8 ]. There is some evidence that multicomponent, non-pharmacological interventions can reduce delirium incidence within in-patient hospital settings [ 9 , 10 ].

Despite the association between delirium and increased mortality rates, extended hospital stays, and long-term impact on overall health, delirium remains underreported and underdiagnosed [ 5 , 11 , 12 ], however there is evidence of recent improvement [ 13 ]. Junior doctors in the United Kingdom (UK) and Ireland have demonstrated a lack of knowledge on how to effectively diagnose and manage delirium [ 14 ]. Furthermore, nurses also possess poor knowledge regarding delirium identification, with difficulties distinguishing between delirium, dementia, and depression reported [ 15 , 16 ]. Therefore, to promote safe and effective care, healthcare professionals need to be equipped with the knowledge and ability to better identify, diagnose and manage delirium. Enhanced proficiency in this area allows for timely interventions and tailored care plans, emphasising the critical role of knowledgeable healthcare providers in optimizing outcomes for individuals with delirium.

Multidisciplinary approaches to delirium education, involving doctors, nurses, and pharmacists learning together, have been shown to be important in improving patient outcomes and learning experiences [ 17 , 18 ]. Implementing multicomponent interventions, such as early mobilisation and family participation, have demonstrated benefits in reducing delirium incidence and duration [ 19 ]. For example, in a community hospital, the formation of an interprofessional consultative Delirium Team improved the prevention, detection, and management of delirium, with two-thirds of referred patients not requiring specialist consultation [ 20 ]. Multidisciplinary approaches to delirium education can therefore enhance patient outcomes and improve the overall learning experiences of healthcare professionals and have been recommended as part of clinical guidelines on the topic [ 2 ].

Interactive education such as role play, games, and simulation, have proven successful in improving qualified healthcare practitioners’ (e.g., nurses, doctors, physiotherapists) awareness of best practice associated with delirium care [ 21 , 22 ]. However, pre-registration delirium education is inconsistent and varies considerably amongst health profession programmes, with approximately only 50% of UK universities providing medical education explicitly on delirium [ 23 ]. Further, in Ireland, limited studies have been identified to uncover what education, if any, is being provided on delirium. Previous studies have aimed to address such issues in undergraduate nursing studies through a face-to-face ‘delirium awareness’ program and podcast [ 24 , 25 ] and through objective structured clinical examinations with medical students leading to improvements in knowledge and self-efficacy on identifying and managing delirium [ 26 ]. However, there remains a dearth of educational resources highlighting the importance of interdisciplinary teamwork in the identification and management of delirium.

Numerous delivery modes exist for educating healthcare professional students. Notably, the digital mode stands out for its success in interdisciplinary student education, fostering interprofessional socialisation, collaborative competencies [ 27 ], creative thinking [ 28 ], and serving as an effective platform for learning about telehealth [ 29 ]. Digital resources also provide a flexible mode of delivery, with participants able to access these in their own time. As not all universities have healthcare disciplines studying concurrently, the flexibility with digital resources may prove beneficial as face-to-face education with all disciplines may be impractical due to timetabling and placement timings. The aim of this study is therefore to co-design and evaluate a digital education resource to improve health profession students’ knowledge and self-efficacy in providing care to patients with delirium. Moreover, this study will involve:

A systematic review to synthesise current evidence on digital delirium education interventions for health profession students.

Focus groups with health profession students to understand their awareness and experiences of providing care to people with delirium.

A series of questionnaires (Delphi study) to determine what key stakeholders perceive as the key education priorities associated with delirium.

Co-design of a digital education resource with health profession students, professionals, and those with lived experience of delirium.

Evaluation of the digital resource to determine usability, perceived usefulness, the user experience, and preliminary efficacy in enhancing health profession students’ knowledge and self-efficacy for providing care to individuals with delirium.

A logic model will be used to provide a systematic guide in the design of the study, including the decision on research outcomes and methods for data collection and analysis [ 30 ]. It is anticipated that the study objectives will be achieved through three key workstreams:

Generating theory.

Co-design of the digital resource.

Intervention evaluation.

An expert reference group (ERG) will be formed prior to study commencement, ensuring the study progresses as intended, that the protocol is adhered to, and to be involved when appropriate to support different elements of the study. The ERG will be comprised of representatives from Northern Ireland and the Republic of Ireland, with personal, professional, and educational knowledge and expertise on delirium. It is envisaged that this will include clinicians, former patients, family carers, policy makers, and educators.

Workstream 1: generating theory

Workstream 1 (WS1) will inform the ‘inputs’ of the logic model. Comprised of three phases, WS1 aims to generate evidence and theory (systematic review and focus groups) and achieve consensus on the key education priorities associated with delirium education for health profession students (Delphi survey). The findings of WS1 will be utilised to inform the initial design and development of the digital education resource.

WS1 phase 1: systematic review

Phase 1 will involve a mixed methods systematic review to synthesise the current evidence on digital delirium education interventions for health profession students. The primary objective of this systematic review is to evaluate how pre-registration healthcare students are equipped to recognise, assess, and implement interventions for delirium prevention through digital or web-based educational interventions. Additionally, the review aims to inform the design of a future digital educational tool for delirium education. It encompasses both qualitative and quantitative studies to scrutinise the impact of existing digital or web-based delirium education programmes on the learning and practice of health professional students in higher education. The study seeks to explore the effectiveness of these digital education programmes, considering various factors such as educational context, professional backgrounds of students, and programme design. The review will also identify facilitators and barriers, assess measures of effect, undertake a critical appraisal assessment, and employ a mixed-methods synthesis approach. Potential subgroups within the broader cohort of health profession students may also be explored. Ultimately, this systematic review will provide comprehensive insights into digital education about delirium, shaping future educational strategies and contributing to enhanced patient care outcomes. A summary of the systematic review protocol is available via PROSPERO: ( https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=422411 ).

The findings of this review will help inform both WS2 and WS3. The review will be conducted in accordance with the Preferred Reporting Items for Systematic reviews and Meta-analyses (PRISMA) checklist [ 31 , 32 ]. The following databases will be searched for eligible studies: CINAHL Complete, Medline, Embase, PsycINFO, Scopus, Web of Science, and Cochrane CENTRAL. Reference lists of relevant systematic reviews and scoping reviews will also be manually searched to ensure additional studies can be identified for inclusion. Studies eligible for inclusion must feature a digital or web-based education intervention on delirium for health profession students in tertiary/third level education, published between 2012 and 2023. The Joanna Briggs Institute Manual for Evidence Synthesis [ 32 ] will inform the integration of qualitative and quantitative data. Quality assessment will be conducted using the Crowe Critical Appraisal Tool (CCAT).

WS1 phase 2: focus groups with health profession students

Face-to-face focus groups will be conducted with health profession students to gain insight into their awareness and experiences of providing care to people with delirium. Approximately 32 health profession students will be recruited from two universities on the Island of Ireland (Queen’s University Belfast and University of Limerick). Participants will be recruited from a variety of health profession programmes, including student doctors, nurses, pharmacists and allied health professions. Students meeting the eligibility criteria will be contacted via email by their Director of Education or course lead, who will provide them with information about the study. These gatekeepers, serving in an independent capacity, will not be directly engaged in the research. Those students expressing interest in joining a focus group can reach out to the research team to receive an information sheet and details regarding the focus group schedule. Prior to participating in the focus group interview, all selected participants will be required to provide written consent. All participants will be reminded that their participation is voluntary and will not impact upon their course grade. Focus groups will be audio recorded and transcribed for analysis.

WS1 phase 3: Delphi survey

Guided by the findings from the systematic review and focus groups, a modified Delphi survey will be conducted to determine what key stakeholders perceive as the main priorities for delirium education for health profession students. It is anticipated that three rounds of surveys will be conducted with key stakeholders. Stakeholders will include individuals, organisations, and communities with a direct interest and/or expertise in health profession education on delirium. The items for the first round of the Delphi survey will be developed using empirical findings from Phases 1 and 2 of WS1, and through input of the research team and the ERG. Identification of delirium experts will be supported through engagement with various organisations including the British Geriatrics Society, the Royal College of Nursing, the All-Ireland Gerontological Nurses Association, the Irish Gerontological Society and other networks known to the research team.

The Delphi survey will be developed online using MS forms and emailed to those participants that have provided informed consent to be contacted via the professional networks. Participants will be asked to answer each item using a Likert scale, with each item requiring at least 75% agreement to proceed to the next round, or, in the case of the final round, be considered to have gained consensus. It is anticipated that 50 delirium experts (25 from each country) will be recruited to take part in the Delphi survey [ 33 ].

Workstream 2: co-design of the digital education resource

Guided by the findings from WS1, workstream 2 (WS2) will focus on the co-design of the digital education resource with key stakeholders. The digital education intervention will form the ‘output’ of the logic model. Co-design of the digital education resource will be conducted with a co-design group, comprised of 15–18 health profession students representing medicine, nursing, pharmacy and allied health. Members of the co-design group will be recruited in the same way as noted in WS1, Phase 2. It is envisaged that four co-design workshops will be held in-person across a three-month period, two in Northern Ireland and two in the Republic of Ireland. To ensure development of the digital resource is evidence-based throughout, findings from WS1 will be incorporated throughout the co-design process, guiding the design, functionality, and key public health messages. The co-design approach proves invaluable for crafting education resources among student populations, as it actively involves them as end-users. This participatory method empowers students to prioritise and shape the content based on their preferences and specific learning needs, ensuring the resulting resources align closely with their educational requirements and enhance overall engagement [ 34 , 35 ].

As part of the co-design process, students will be regarded as experts whose insights will inform critical decisions regarding the content of the digital education resource. One key aspect that will be explored is whether all healthcare professionals require uniform knowledge about delirium or if tailored sections specific to their roles are necessary. For instance, doctors may prioritise understanding the pathophysiology of delirium, while pharmacists might focus on medications and polypharmacy, and nurses may emphasise initial symptom recognition. This iterative approach acknowledges the diverse learning needs within healthcare disciplines and recognises that preferences may vary. By integrating empirical evidence from workstream 1, including a systematic review, qualitative focus groups, Delphi survey, and the co-design methodology itself, the development process will remain evidence-based and responsive to the evolving needs of the end-users.

Workstream 3: evaluation

WS3 will involve the mixed methods evaluation of the digital resource with health profession students through two phases, producing the ‘outcomes’ of the logic model. This process will be guided by the technology acceptance model (TAM) [ 36 ] and Proctor et al.’s [ 37 ] taxonomy of implementation outcomes. Originating in 1986, the TAM aids in understanding predictors of human behaviour regarding acceptance or rejection of technology through two variables that may affect the adoption of digital technology: 1) perceived usefulness and 2) perceived ease of use.

These variables, and students’ usage patterns, will be assessed as part of the intervention evaluation. The components of Proctor et al.’s taxonomy of implementation outcomes that will underpin the evaluation are:

Acceptability

The perception among stakeholders that the given treatment, service, practice, or innovation is agreeable, palatable, or satisfactory.

The intention, initial decision, or action to try or employ an innovation or evidence-based practice (uptake) .

Appropriateness

The perceived fit, relevance, or compatibility of the innovation for a given practice setting, provider, or consumer; and/or perceived fit of the innovation to address a particular issue or problem.

WS3 phase 1: health profession students’ knowledge, self-efficacy, and usability

Phase 1 will involve the assessment of health profession students’ knowledge and self-efficacy of providing care to those with delirium through a pre- and post-test questionnaire. Two questionnaires, the 35-item Delirium Knowledge Questionnaire (DKQ) [ 38 , 39 ] and a 3-item questionnaire on self-efficacy towards recognising and providing care to those with delirium [ 24 , 25 ]. Questionnaires will be delivered to participants at baseline and four weeks after the delivery of the intervention.

Approximately 300 health profession students from both Queen’s University Belfast and University of Limerick will be recruited through convenience sampling in the same matter noted in WS1, Phase 2 and WS2. Students will be provided with an opportunity to an online information sheet detailing the study and how to use the resource. A series of questions will be displayed to gain consent, with access to the questionnaire granted only once consent is provided. Usability of the digital resource will be assessed through the validated 10-item questionnaire, the ‘System Usability Scale’ [ 40 ] provided post-test. Lastly, participants will be provided with two ‘open text’ questions to facilitate additional comments on what students liked or disliked about the resource, and to suggest ideas for future dissemination or testing activities. Internal analytics such as user engagement and time spent on the resource will be used to further determine usability and acceptability.

WS3 phase 2: perceived ease of use and usefulness of the digital resource

The second and final phase of the evaluation will aim to uncover health profession students’ perceptions regarding the ease of use and usefulness of the digital resource. Four focus groups will be conducted with health profession students ( n  = 32) who have previously used the resource. These participants will be asked to provide consent to be contacted for this part of the evaluation after they complete WS3, Phase 1. Dependant on data saturation, the number of participants may be either increased or decreased. Focus group questions will be developed by the research team, aligned with the RE-AIM (Reach, Effectiveness, Adoption, Implementation, Maintenance) framework [ 41 ] and Proctor et al.’s taxonomy on implementation outcomes [ 37 ]. Students will be asked to share suggestions on approaches for the resource to reach a wider student audience, how they would identify the effectiveness of the resource, ways in which the resource could be adopted and implemented by healthcare and educational institutions, and how it should be maintained over its lifetime.

Data analysis

Qualitative analysis.

Audio recordings and notes gathered through the focus groups in WS1 and WS3 will be transcribed verbatim and uploaded to NVivo 12 management software for analysis, along with the ‘open text’ comments from the post-test questionnaire (WS3) and those gathered during the co-design workshops (WS2). Qualitative data will be coded and analysed using thematic or narrative synthesis (systematic review), constant comparative analysis (WS1 focus groups), and thematic or directed qualitative content analysis (WS2 and WS3 focus groups).

Quantitative analysis

This will involve quantitative data from the review’s data extraction form (WS1) and evaluation data from the pre-post questionnaires (WS3). Quantitative data analysis will be conducted using SPSS v.26. Descriptive statistics will be employed to report on results of the Delphi study, participant demographics, and internal analytics. Paired t-tests will be used to assess changes to health profession students’ knowledge of delirium and self-efficacy to determine preliminary efficacy. Additionally, demographic details will be collected to gauge which participants engage with the resource (providing data on its reach).

The implementation of an online platform for the delivery of the co-designed digital education resource will not only address the pressing need for interprofessional education on delirium care but also has advantages associated with digital learning environments. Despite a scarcity of literature specifically addressing interprofessional digital education resources on delirium and their impact on delirium knowledge, self-efficacy, and healthcare practice, existing studies examining the effects of digital education interventions on professional knowledge and self-efficacy offer important findings. Research has shown that digital education interventions can significantly enhance learning outcomes for both health profession students and registered professionals, providing an engaging and interactive learning experience [ 42 , 43 , 44 , 45 , 46 , 47 ]. By adopting an immersive approach with real-time feedback, digital interventions have the potential to boost students’ confidence and self-efficacy in their knowledge and skills [ 44 , 45 , 46 ].

One of the key advantages of digital interventions is their superior accessibility compared to traditional classroom-based teaching methods, particularly in the context of interdisciplinary education. This accessibility ensures that students from various healthcare disciplines, with differing schedules and logistical constraints, can access essential information about delirium care at their own convenience [ 48 ]. Moreover, the asynchronous nature of digital resources facilitates flexible learning, allowing students to progress through the material at their own pace and revisit key concepts as needed. This flexibility is particularly advantageous given the complexities of coordinating educational sessions involving nurses, doctors, and pharmacists, who may have disparate timetables and program requirements.

While the e-resource will serve as a valuable adjunct to traditional educational methods, it is essential to recognise that it will not be designed to replace simulation, practice-based learning, or face-to-face teaching that may already be in place within a healthcare professional programme. Rather, the aim is to complement existing educational practices by providing a comprehensive overview of delirium care and promoting interdisciplinary collaboration among healthcare professionals.

Limitations and challenges

There are several limitations to consider in this study. First, there is a risk of response bias through the pre-post questionnaires (WS3) due to self-reporting which may not reflect the true impact of the digital resource on knowledge and self-efficacy. However, the utilisation of validated questionnaires should minimise this limitation. Second, as the resource will be developed in the context of health profession students on the island of Ireland, the generalisability of the resource to other contexts and populations of health profession students may pose a limitation. However, similar standards and competencies are expected for undergraduate health profession programmes nationally and internationally, it is expected that the digital resource will have reach and the potential to be adapted for different cultural contexts.

Lastly, there are potential challenges in the recruitment and retention of health profession students throughout different stages of their education programme. Thus, findings of this study may not be representative of the learning needs and preferences of all health profession students. However, through integrating local, national, and international evidence, and gaining insight from experts and those with lived experience of delirium, it is expected that the resource will be an engaging and evidenced-based resource that is acceptable to end-users.

Data availability

No datasets were generated or analysed during the current study.

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Acknowledgements

This research project, DelHPIre (Delirium Health Professionals Ireland), is funded as a ‘Strand I Project’ by the Shared Island North–South Research Programme (from the Department of Further and Higher Education, Research, Innovation and Science in the Government of Ireland) to improve the prevention, recognition, and management of delirium across the island of Ireland. Reference: 6692- HEA North South.

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All authors have agreed the final version of this study protocol and agree to be accountable for this content. All authors have also met criteria adapted from McNutt et al. Proceedings of the National Academy of Sciences, Feb 2018, 201715374; DOI: https://doi.org/10.1073/pnas.1715374115 ; licensed under CC BY 4.0. All authors been involved in substantial contributions to theconception (GM, AC, CBW, PB, PS, MG, DT, HB, JM, MB, AT, AMc), design of the work (LC, AC, CBW, PB, PS, MG, JMc, DT, HB, JM, MB, AT, TA, AMc, EC, GC, GM); have drafted the work or substantively revised it (LC, AC, CBW, PB, PS, MG, JMc, DT, HB, JM, MB, AT, TA, AMc, EC, GC, GM). The authors read and approved the final manuscript (LC, AC, CBW, PB, PS, MG, JMc, DT, HB, JM, MB, AT, TA, AMc, EC, GC, GM).

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This study has received ethical approval from the Faculty of Medicine, Health & Life Sciences, Queen’s University Belfast (MHLS23_50, MHLS23_140, MHLS23_122) and the Faculty of Education & Health Sciences, University of Limerick (2023_01_02_EHS, 2023_06_20_EHS). Written consent will be gathered via email or hardcopy consent sheets. Where this is not possible, an audio recording of verbal consent will be obtained. All student participants will provide informed consent to participate in the study. Participant information sheets will be provided to those who express an interest in taking part in the study, and those formally recruited reassured that they have the right to withdraw from the study at any time.

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Cook, L., Coffey, A., Brown Wilson, C. et al. Co-design and mixed methods evaluation of an interdisciplinary digital resource for undergraduate health profession students to improve the prevention, recognition, and management of delirium in Ireland: a study protocol. BMC Med Educ 24 , 475 (2024). https://doi.org/10.1186/s12909-024-05468-1

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Exploring patients’ perspectives: a mixed methods study on Outpatient Parenteral Antimicrobial Therapy (OPAT) experiences

• Sophie Peter 1 , 2 ,
• Charlotte Oberröhrmann 2 ,
• Holger Pfaff 3 , 4 ,
• Clara Lehmann 5 , 6 , 7 ,
• Kirsten Schmidt-Hellerau 5 ,
• Vanessa Brandes 5 ,
• Charlotte Leisse 5 ,
• Christoph Heinrich Lindemann 8 ,
• Peter Ihle 9 ,
• Jutta Küpper-Nybelen 9 ,
• Anna Hagemeier 10 &
• Nadine Scholten 2  

BMC Health Services Research volume  24 , Article number:  544 ( 2024 ) Cite this article

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Metrics details

Outpatient Parenteral Antimicrobial Therapy (OPAT), an alternative to inpatient intravenous antibiotic therapy, has shown benefits in international studies such as increased patient satisfaction. Because OPAT has been used only sporadically in Germany so far, no structured results on patients’ experiences and concerns regarding OPAT have yet been available. This study therefore aims to explore the experiences of OPAT patients in a pilot region in Germany.

This is an observational study in a German pilot region, including a survey of 58 patients on their experiences with OPAT, and in-depth interviews with 12 patients (explanatory-sequential mixed-methods design).

Patients reported that they were satisfied with OPAT. That a hospital discharge was possible and anti-infective therapy could be continued in the home environment was rated as being particularly positive. In the beginning, many patients in the interviews were unsure about being able to administer the antibiotic therapy at home on their own. However, healthcare providers (doctors and pharmacy service provider staff) were able to allay these concerns. Patients appreciated regular contact with care providers. There were suggestions for improvement, particularly concerning the organization of the weekly check-up appointments and the provision of information about OPAT.

Conclusions

Patients were generally satisfied with OPAT. However, the treatment structures in Germany still need to be expanded to ensure comprehensive and high-quality OPAT care.

Trial registration

NCT04002453, https://www.clinicaltrials.gov/ , (registration date: 2019–06-21).

Peer Review reports

When hospitalisation is required solely to administer intravenous antibiotic therapy to treat an infectious disease, Outpatient Parenteral Antimicrobial Therapy (OPAT) offers a means to entirely circumvent hospitalisation or at least significantly reduce its duration [ 1 , 2 ]. OPAT involves the delivery of approved parenteral antibiotic therapy outside of an inpatient hospital setting. Typically, a secure vascular catheter is inserted, allowing for the administration of the anti-infective therapy via infusion. OPAT can be carried out in various settings, including a general practitioner’s office, a specialised outpatient facility, or even the patient's own home [ 2 ]. Patients may self-administer or receive assistance from an informal caregiver or professional caregiver [ 2 , 3 ]. Typical indications for OPAT include skin and soft tissue infection, bone infection, and endocarditis, which are often associated with the need for prolonged intravenous antibiotic therapy [ 2 , 4 ]. Offering comparable efficacy to inpatient care, OPAT presents numerous advantages, including a lower risk of nosocomial infections [ 5 , 6 ]. At the same time, costs can be saved by preventing or shortening hospital stays [ 4 , 7 , 8 , 9 ]. OPAT is therefore an efficacious and safe alternative to inpatient treatment [ 10 , 11 ]. Research on OPAT consistently underscores the high levels of patient satisfaction attributable to its seamless integration into their daily routines [ 8 , 12 , 13 , 14 , 15 , 16 ]. OPAT is a standard care practice in many countries [ 3 , 17 , 18 ]. However, in Germany, there is a lack of healthcare infrastructure, regulations, and recommendations to support OPAT [ 1 ]. As a result, OPAT is only offered by few specialised centres or practices [ 2 , 19 ]. There has been little research about patients’ experiences with OPAT in Germany, despite the fundamental and active role of patients in this mode of treatment. In the spirit of patient-centred outcome measurement, this article aims to answer the question: How is OPAT experienced and accepted by patients in an urban pilot region of Germany (Cologne metropolitan region)? In a nationwide comparison, Cologne has an advantage in the developement of an OPAT structures: an infectiological network (consisting, for example, of hospitals and outpatient infectiological practices) dedicated to patient care, education and training and also research, e.g. for OPAT [ 1 ].

A prospective observational study as part of the K-APAT study (“outpatient parenteral antibiotic treatment in the metropolitan region of Cologne”), which scientifically evaluated the implementation of OPAT in a German pilot region (the Cologne metropolitan region), was conducted. Clinical data is published separately [ 20 ]. A detailed description of the study design can be found in the study protocol [ 21 ]. The study has been approved by the Institutional Review Board of the University of Cologne, Germany (19–1284-1). The study is a multi-centre study consisting of 5 hospitals and 5 outpatient practices with a focus on infectiology. All study centres were allowed to include patients in the study [ 21 ]. Patients who were considered suitable for OPAT by the infectious disease service were also enrolled in the patient satisfaction survey study after giving informed consent to OPAT. Inclusion criteria were therefore admission as an OPAT patient; a minimum age of 18 years; and written informed consent to the survey study. As the survey documents could only be provided in German, sufficient knowledge of German was mandatory. Data collection was carried out using an explanatory mixed-methods design between November 2019 and September 2021 [ 22 ]. The explanatory mixed-methods design allows a broad yet thorough understanding of the patient perspective [ 22 ]. Patients were invited to answer up to three surveys delivered by mail about their OPAT experience at three timepoints: T0 (before starting OPAT, 48 items), T1 (two weeks after starting OPAT, 51 items) and T2 (one week after finishing OPAT, 42 items). Most of the items were likert scaled. All questionnaires included one open ended question (“If you have any comments, please note them here:”). After the patients were enrolled in the study, a written questionnaire was sent to their homes if they had given their consent. The data was collected pseudonymously in returning the questionnaire to the research institute, which was not involved in the treatment of the patients. The questionnaires were developed based on the current literature [ 2 , 11 , 13 , 23 , 24 , 25 , 26 , 27 , 28 , 29 ] and critically reviewed by the study team consisting of social scientists, healthcare researchers, and clinicians with expertise in infectious diseases. The questionnaires used in the K-APAT study were developed specifically for this research. A German version is available in the project's final report [ 30 ]. Both self-developed questions and validated instruments were used in the questionnaires. The final report also contains information on validated and self-developed items [ 30 ]. The questionnaires were tested for comprehensibility and adapted within the framework of ten cognitive pretests with healthy adult persons of different age groups (19 to 70 years old). Topics of the questionnaires are shown in Table  1 (Tab. 1 ). See the appendix for the English version of the questionnaire items used for this publication (Additional file 1 ). Data were analysed descriptively using Stata 17 software.

After the quantitative data collection, in-depth interviews were conducted with selected patients who answered the questionnaires beforehand. These patients had indicated in the written declaration of consent for the study that they would be interested in taking part in in-depth interviews once the questionnaire survey had been completed. The patients were selected for the interviews by the researchers using the pseudonymised data set. The researchers were not involved in the patients' medical care and were only familiar with the patients’ T0, T1 and T2 questionnaires. The researchers wanted to map the greatest possible variability of the patients, especially with regard to their experiences and satisfaction with OPAT. Patients were also selected for a balance in gender, age, duration of OPAT treatment, and overall satisfaction with OPAT. All 12 interviews took place in person (mostly at patients' homes) or by telephone, due to contact restrictions during the COVID-19 pandemic.

Topics of the interview guide are shown in Fig.  1 (Fig.  1 ). The English version of the interview guide is available as a Supplementary file (Additional file 2 ). Dimensions of the semi-structured interview guide were developed from the literature [ 11 , 23 , 24 , 25 , 26 , 27 , 28 , 29 ] and expert feedback. The interview guide utilised in the K-APAT study was specifically developed for this research. The German version can be found in the final report [ 30 ].

Topics of the interview guide

The qualitative data were interpreted by means of a content analysis (inductive and deductive categories), which was carried out iteratively by SP and CO [ 31 ].

The participants received a financial incentive for completing the questionnaires and participating in the interviews.

Patients’ characteristics can be found in Table  2 (Tab. 2).

As stated in the publication on the projects’ clinical data, the most frequently treated infections were joint and bone infections (26% of the patients) and vertebral osteomyelitis (14% of the patients) [ 20 ]. All results (including the overall results of the three questionnaires and the interviews) and all items of the survey instruments are included in the final report of the K-APAT project [ 30 ].

Results from the questionnaires

All 77 patients included in the study returned their questionnaires (response rate: 100%). 19 respondents did not answer at least one item of the questionnaire which are reported here. These patients were therefore excluded from the following analyses.

In the first and second questionnaire (T0 and T1) patients were asked about their experiences with OPAT subdivided in e.g. therapy confidence, experiences with intravenous antbiotic therapy and patient education (all topics are summarised in Table  1 ). In T2, all patients were asked to summarise their OPAT treatment. The aim was to obtain a final evaluation of their entire OPAT treatment. All respondents had a positive opinion on OPAT (Fig.  2 ).

Opinions on OPAT

Also as part of the final conclusion to their OPAT most patients rated the organisation of their treatment as good (29.3%) or very good (70.7%) (Fig.  3 ).

Rating of treatment organisation

The majority of patients (98.28%) would choose OPAT again if required. A similarly high proportion (96.55%) would recommend OPAT (Figs. 4 & 5 ).

“Would you choose OPAT again if you suffered from another disease that could be treated with OPAT?”

“Would you recommend OPAT to other patients suffering from diseases that can be treated with OPAT?”

Patients were asked to indicate their problems with OPAT using a Likert scale (strongly disagree, rather disagree, rather agree, strongly agree): Problems with material storage were infrequently reported, with only 10.34% strongly agreeing and 3.45% rather agreeing. A small number of individuals (1.72% strongly agree, 8.62% rather agree) reported feeling uncomfortable with the vascular catheter, while 13.9% tended to agree that the vascular catheter affected how they were perceived in public. Additionally, 12.07% strongly agreed and 18.97% rather agreed that they deliberately hid their vascular catheter in public. Eight people (3.45% strongly agree, 10.34% rather agree) reported complications during their treatment. None of the respondents reported any mistakes made by the medical staff, or that OPAT was an inappropriate treatment option for them (Table  3 ).

Results from the interviews

The results of the interviews are summarised in the Table  4 (Tab. 4).

The interviews lasted between 20 and 58 min (mean length: 39 min).

Despite the overall positive evaluation of OPAT, some difficulties were identified during the interviews following explicit enquiries. Eight of the interviewees reported anxiety or uncertainty, especially at the beginning of their therapy (e.g., fear of making mistakes when administering antibiotics; fear of venous catheter management because of the “at heart” location). One patient commented: “This [OPAT] is excellent. Painless and great. However, it is a strange feeling considering the access is in the heart [sic]. (…) You do have a queasy feeling. (…) But it is great. I always thought: Well, what if it slips? But how is it supposed to slip? Slipping is not possible.” (patient 4). However, most fears quickly subsided after the start of therapy. One patient reported severe mechanical problems due to blockage of the catheter, which led to her being switched to oral medication for the last 3 days of her treatment: "Overall it went well and only few problems occurred. Sometimes it happened that, [patient paused and reformulated] So once I had difficulty taking it [the pump system] off again. We then had to use a pipe wrench.” (patient 8). However, most patients had no problems with the OPAT at all. One interviewee said: “And, yes, here at home I had no problems at all. Even the first application, I did it a bit more carefully, of course, just to not forget anything, to do everything right, and very quickly routine comes in.” (patient 5). Another patient summarised: “There were no problems of any kind. There was no pain. There were no incidents.” (patient 9). Motor and cognitive limitations were mentioned as possible barriers to handling infusions.

The interviewees reported numerous benefits of the OPAT. They could be discharged from the hospital early, which they associated with greater self-determination and higher quality of life than in the hospital. One patient stated: “So that [the OPAT] has already given me a lot of freedom.” (patient 11) and another patient mentioned: “As I said, I’m at home, I have my familiar surroundings, and then I feel like I’m back to a normal life and I have the disease under control. So it [the OPAT] was a good way to not have to keep going back to the doctor again and again.” (patient 6). All interviewees emphasised how important it was for their well-being to be at home. One patient explained: “That I can be at home. That’s important for me because I feel most comfortable at home. I have 100 TV channels, and at the hospital, I have five that I don’t really watch at home. Or I can eat whatever I feel like. Home is home.” (patient 7). Freedom and independence in a homecare setting were highly valued compared to the lack of freedom and dependence in inpatient care. A lot of patients explained that OPAT at home was easy to perform and helpful for recovery. In addition, the treatment was predictable: “That was ritualised. I then took a book at some point and read, sat down in the corner and read. I didn't move wildly, right?” (patient 3). Patients did not feel that OPAT interfered with their daily life. In the words of one interviewee:”So everything I do here at home, I’ve been able to do with the [venous catheter].” (patient 10). One patient added: “I would say top-notch. I can only recommend it to everyone. So who is fit enough: definitely. Yes. Although I was also in the hospital [patient paused and started a new sentence] They were all super nice, doctors, nurses, everyone. They really did their best, but home is home, right? And if you really have the opportunity, (…) immediately. So I would do that again immediately. Yes. I hope not that I need it again, but let’s just say I would do it in a heartbeat.” (patient 12). In addition, three patients who work in healthcare themselves stated that OPAT is associated with cost savings for the healthcare system. They emphasised that no hospitalisation or nursing service is required for this treatment. From their experience, the venous catheter can stay in place for longer and does not need to be constantly replaced compared to peripheral catheters, and they heal well.

The interviewees reported some restrictions in everyday life due to OPAT: e.g., sleeping on the side with the catheter is uncomfortable, financial challenges (e.g., co-payment for those with statutory insurance was not explained and cost coverage for those with private insurance was initially unclear, both should be communicated more clearly), insecurity in handling, and suspected side effects (e.g., fatigue, eczema, exhaustion that patients attributed to their OPAT) were also reported by the patients. Most patients did not describe any side effects: “And I didn’t have any side effects or anything. So not that I felt anything bad, that I felt bad, that I felt dizzy or anything. It went wonderfully.” (patient 1).

The interviewees also reported their experiences with healthcare providers. Often patients mentioned a lack of awareness of OPAT among healthcare providers in the hospital and organisational barriers such as waiting times. The venous catheter placement and removal took place in the hospital for all patients. Weekly check-ups were also primarily performed in a hospital outpatient clinic. One patient reported on her hospital stay as: “I already noticed that the doctor was a bit worried […] and then [he] told me to make sure that I observe the hygiene measures, that would be very important. I think there is a bit of fear when you hand things over to the patient: let's see what the patient does with it himself? Surely there are differences there, right?” (patient 8). The interviewed patients evaluated the support by the pharmacy service providers as well-organised. The deliveries of the material (e.g. bandages, material for cleaning catheters, disinfectant) were organised well and without problems. The study participants received a high quantity of material, often more than was required. The OPAT briefing was good and adapted to the patients' needs. Nevertheless, the interviewees reported that the information they got from their healthcare providers was partially incomprehensible (e.g. the step-by-step instruction of the OPAT is very complex with preparation of the material, cleaning of the skin and the catheter and connecting the pumps). One patient felt that she had generally received too little information about OPAT from her physicians. The patients liked the check-ups and the option to contact the healthcare providers by phone: “Yes, she [the pharmacy employee] also called me again herself, I think on the fourth day, which I thought was very nice, and asked if everything was okay, if I was getting along, if I needed anything else.” (patient 2). Outpatient providers such as General Practitioners and other outpatient care services played a minor role among respondents and were only sporadically involved in treatment. Overall, the patients rated the organisation of the OPAT as good.

Main findings

In the K-APAT study, a prospective observational study, we aimed to investigate medical care with OPAT, focusing on the model region of Cologne. The goal was to assess the feasibility and success of implementing OPAT within the German healthcare system. The sub-study we presented here aimed to find out more about patient views on OPAT, their satisfaction, and their experience within the framework of a mixed-methods study.

In our study, OPAT had high levels of patient satisfaction, with nearly all patients expressing contentment. Other studies found comparable satisfaction with OPAT treatment with similar rates of recommendation and reporting that they would opt for OPAT again if necessary [ 13 , 26 ]. Saillen et al. (2017) concluded that OPAT patients were “happy to take over some responsibility for their treatment” [ 26 ]. In general, patient satisfaction with OPAT is comparably high internationally [ 8 , 12 , 13 , 14 , 15 , 16 ]. Quintens et al. (2020) also reported that all respondents were very satisfied with their OPAT treatment [ 32 ]. However, some patients in their study as well as in Berrevoets et al. (2018) also wanted more information about the OPAT and reported side effects attibuted to the OPAT [ 11 , 32 ] as the patients in our interviews did too. Similarly as can be seen in our data, Saillen et al. (2017) reported that patients initially had concerns about the self-application of and reported on mechanical problems in handling the pump system [ 26 ].

Notably, the most satisfying aspect was that this form of treatment allowed patients to be discharged from the hospital. This effect does not only apply to the OPAT patients reported here: As hospitalisation is often perceived as a burden [ 33 , 34 ], a lot of patients express satisfaction upon discharge from the hospital. The organisation of OPAT was largely perceived to be good. In particular, patients appreciated regular contact with healthcare providers (physicians and pharmacy service providers). It is important for the success of the therapy that patients feel comfortable and well cared for [ 11 ].

In countries where OPAT is well established, the number of patients participating in OPAT studies is significantly higher [ 35 ]. One Italian study showed that the COVID-19 pandemic supported the use of OPAT [ 10 ]. The number of participants in our study fell during the pandemic. We suspect that this is due to the heavy burden on hospital staff and outpatient practices.

Strengths and limitations

It is important to consider limitations when interpreting the results of this study. It was conducted in the densely populated metropolitan region of Cologne: the findings may therefore not be fully representative of other regions in Germany (e.g. rural areas, regions without infectiological networks). But the region was well suited to conducting a feasibility study on OPAT due to the already established expertise of the regional, infectiological network. In addition, most of the study was conducted during the COVID-19 pandemic, which could have led to a number of limitations. For example, our participant number ( N  = 58) is low. Although all 77 patients we included in the study returned their questionnaires (response rate: 100%), due to missing values, we had to remove 19 patients from the dataset. We attempted to improve recruitment by having multiple study sites and conducting the study in a region that has a good OPAT care structure by national standards. Nevertheless, the case number limits the generalisability of the data. However, other international studies have reported similarly low participation rates [ 32 , 36 , 37 , 38 ].

Compared to other studies our patient population was mostly younger (mean of survey patients: 55.2 years) except for the study presented by Al Shareef et al. (2022) with the same median age as in our study [ 38 ]: Chambers et al. (2019) reported on patients with a median age of 61 years [ 35 ]. Saillen et al. (2017), Staples et al. (2022) and Berrevoets et al. (2018) also reported a higher median age of 59 years, 62 years and even 68 years [ 11 , 14 , 26 ]. Treatment periods in our population (survey data: mean: 15 days, interview data: mean: 27 days) were higher than in other studies. Wolter et al. (2004) reported about 11 days, Hase et al. (2020) reported 13 days and Saillen et al. (2017) 8.5.days [ 8 , 26 , 36 ]. As is common in many other OPAT studies, our survey data also has a high proportion of male participants. [ 11 , 14 , 32 , 36 , 38 ].

It is particularly interesting that in the survey, OPAT was described as good or very good. More detailed insights from the interviews showed that, despite the overall good rating, patients had minor problems with OPAT. Thus, the mixed-methods design and the extensive interview data complement the quantitative data well, e.g. patients were able to report on their experiences in more detail and more individually than it was possible in the questionnaires. Thus, the mixed-methods design supported the breadth and depth of data [ 39 ] on patients' OPAT experiences.

We tried to include a wide range of patient experiences with OPAT in the in-depth interviews and have made efforts to achieve a gender and age balance of the interviewees. But as you can see in Table  2 the interviewees were a little older and had a longer treatment period of OPAT compared to the survey population. In addition, the proportion of men in the survey was higher than in the interviews. Despricption on the underlying diseases can be found in the publication on the clinical data [ 20 ]. Unfortunately, patients who would not recommend OPAT ( N  = 2) to others or tended to be less satisfied than average either did not consent to be contacted for an interview or were unwilling to be interviewed when asked to do so.

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Implications

In the long term, OPAT can be a suitable option for standard care.

OPAT not only increases patient satisfaction through cross-sector, indication-based and patient-oriented care, but also relieves the burden on hospitals by reducing inpatient bed days [ 1 ]. Moreover, the inpatient sector is facing mounting pressures arising from a scarcity of specialists, demographic shifts, and soaring costs. In response, healthcare policy is advocating for the expansion of the outpatient care model, guided by the principle of “outpatient care before inpatient care” [ 40 ].

The data has shown that the OPAT programme can be improved in the following ways to further increase patient satisfaction: Some patients reported they did not know about potential co-payments, which should be communicated more clearly. Waiting times for check-ups in hospitals or doctors' offices should also be reduced. Patients have expressed the wish for flyers or information material explaining the OPAT procedure as well as the handling of the intravenous access in more detail. This improvement has already been introduced into care at the study sites: Flyers and videos describing the OPAT procedure have been developed.

This study shows a high level of patient satisfaction with OPAT. The preference for home-based treatment over hospital care is a key contributing factor. OPAT is still a relatively uncommon procedure in Germany, which is why there are still no nationwide structures for this treatment option. However, our studie shows that patients are satisfied with the care they received and that an expansion of the OPAT structures should therefore be considered. In the studied region, a quality infrastructure for OPAT exists, marked by specialists in specialised and interconnected outpatient clinics. Due to all of these advantages, it is important to further promote the use of OPAT and broaden these prerequisites by establishing adequate OPAT structures [ 41 ].

Availability of data and materials

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

Abbreviations

Outpatient parenteral antibiotic treatment in the metropolitan region of Cologne

• Outpatient Parenteral Antimicrobial Therapy

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Acknowledgements

We would like to thank all patients who participated in our study. We would also like to thank all of the staff at the study centres and the pharmacy service providers who cared for the patients together with the study physicians.

Open Access funding enabled and organized by Projekt DEAL. This work was supported by the German Federal Joined Committee (grant number 01VSF18036).

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Sophie Peter, Charlotte Oberröhrmann & Nadine Scholten

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ClL led the K-APAT study. NS, CO and SP designed the questionnaires and the interview guide, and have mainly carried out and evaluated the data collection. SP wrote the first draft of the manuscript. AH, ChL, ClL, CO, HP, JKN, KSH, NS, PI and VB substantively revised the work. All authors read and approved the final manuscript.

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Peter, S., Oberröhrmann, C., Pfaff, H. et al. Exploring patients’ perspectives: a mixed methods study on Outpatient Parenteral Antimicrobial Therapy (OPAT) experiences. BMC Health Serv Res 24 , 544 (2024). https://doi.org/10.1186/s12913-024-11017-9

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REVIEW article

Current management strategies of urachal anomalies in pediatric patients: a scoping review.

• 1 College of Medicine, University of Central Florida, Orlando, FL, United States
• 2 Division of Urology, Nemours Children's Health System/Nemours Children's Hospital, Orlando, FL, United States

Introduction: Management of urachal anomalies in pediatric patients has historically lacked a clear consensus between conservative and surgical management. We aimed to review and summarize the literature on the diagnosis, symptoms, and evolution in the management of urachal anomalies in pediatric patients.

Methods: We performed a scoping literature review of PubMed/Medline and WebOfScience from January 2000 to February 2022.

Results: 32 publications were selected for inclusion in this analysis. 1,438 unique studies were identified with 32 studies meeting inclusion criteria. 15/32 studies discussed both conservative and surgical management, 14/32 studies discussed only surgical management outcomes, and 3/32 studies discussed diagnostic methods. The studies discussing conservative management supported the treatment of urachal anomalies with an initial conservative approach, which includes watchful waiting, repeated ultrasounds, lesion measurement, and antibiotic use. 5/32 of the included studies identified patients that were converted from conservative to surgical management with conversion rates ranging from 12.5% to 43.5% per study. 14/20 converted patients were identified to have a urachal cyst and 13/20 had a persistent infection.

Conclusions: Strong evidence exists that supports initial conservative management over surgical management of pediatric urachal anomalies. However, predictive factors for determining which patients will require surgical management remain elusive. Treatment algorithms can potentially be developed once carefully developed prospective studies delineate statistically significant patient factors which necessitate surgical management over observation.

Introduction

The urachus is a connection between the early fetal bladder and the allantois that aids in the removal of nitrogenous waste through the umbilical cord and placenta during gestation. This connection typically obliterates during fetal development or early in the neonatal course, forming the median umbilical ligament ( 1 , 2 ). This lumen may fail to obliterate, thus leading to the abnormal persistence of an embryonic urachal remnant. Urachal remnants can be subtyped into different morphological anomalies including a patent urachus which has a fully patent lumen between the bladder and umbilicus, a vesicourachal diverticulum which contains a blind pouch attached to the bladder, a urachal cyst which contains a small patent lumen with two closed ends, and an umbilical-urachal sinus which contains a patent lumen at the umbilicus but does not extend to the bladder ( Figure 1 ) ( 3 ). These four subtypes are classically reported in the literature, however, they may be further subdivided into anatomical variants based on length and connection with the lateral umbilical ligaments ( 4 ).

Figure 1 Illustration of a normal fully developed bladder with urachus and four of the most common urachal anomaly subtypes including patent urachus, vesicourachal diverticulum, urachal cyst, and umbilical-urachal sinus.

Urachal anomalies (UAs) were historically thought to be a rare occurrence, but modern increased utilization of imaging studies has revealed that UAs are more common than previously recorded. In 2015, Gleason et al. reported a 1.03% prevalence in the general pediatric population ( 5 ). Urachal malformations have varied clinical presentations, from asymptomatic (as the most common) to presenting with multiple non-specific symptoms including umbilical leakage, fever, abdominal pain, or acute/recurrent infection of the structure ( 6 ). Current management strategies for UAs include non-operative, conservative management in which the patient is followed to ensure symptoms do not worsen or recur, as well as surgical management in which the structure is removed, either in an open, laparoscopic, or robotically-assisted laparoscopic approach. The lack of a clear consensus on preferred management continues to persist due to the rarity of these defects and a paucity of existing data. Therefore, both approaches continue to be utilized. In this study, we aim to review the literature and identify current management strategies for pediatric patients with UAs.

Materials and methods

Search strategy.

The following keywords were used to search the entire PubMed database on our search date of February 29, 2022, as follows: (urachal remnant) OR (urachal anomaly) OR (urachal abnormality) OR (patent urachus) OR (urachal diverticulum) OR (urachal sinus) OR (urachal cyst); these search terms were adjusted for use in Web of Science. Duplicates, articles published before 2000, and studies published in non-English languages were then removed using automated PubMed and WebOfScience filters.

Inclusion and exclusion criteria

Published original research was analyzed to evaluate the diagnosis and management of UAs in pediatric patients. Studies were included if they contained a group of pediatric patients with a diagnosed UA and data available on patient age, diagnostic techniques, and management. To focus on more recent management strategies, only articles published during or after the year 2000 were included. The abstract and title of the remaining studies were screened by two medical student independent reviewers (YG and DG) and the decision to include and exclude studies was taken in consensus with pediatric urology faculty. Figure 2 summarizes the article identification process. Studies were excluded if they fell into the following categories: animal studies, case reports, adult population included (>18 years of age), reviews, lack of abstract and full text, non-primary research (i.e., systematic review, literature reviews, etc.), and articles not primarily focused on UAs and their management. All studies that met the inclusion criteria were utilized to minimize study selection bias. Articles that remained were read in their entirety.

Figure 2 Flowchart of identification, screening, eligibility, and inclusion of studies in final analysis.

Study identification and characteristics

Of the 1,418 unique studies identified through the initial literature search, 32 were ultimately included in this review as described in Figure 2 along with reasons for exclusion. The characteristics of each individual study are listed in Appendix 1 including the study type, number of patients, diagnostic imaging, type of UA present (if specified), and management strategy. These characteristics are summarized in Table 1 .

Table 1 Characteristics of included studies and patient population.

Characteristics of the included studies, including study type, are noted in Table 1 ( 1 , 5 , 7 – 36 ). One of the retrospective chart reviews also contained elements of a prospective surgical study where 8 patients were randomly assigned to receive surgical treatment ( 1 ). Three of the studies (9.4%) classified as retrospective chart reviews discussed diagnostic methods and reviewed imaging studies used in diagnosis without primarily focusing on the treatment of UAs ( 8 , 25 , 31 ).

Of the 32 included studies, 18 of them (56.3%) reviewed patients who were only treated surgically and discussed variations in surgical technique and management, while 14 of them (43.8%) reviewed patients who received either conservative, surgical, or combined management. None of the studies reviewed solely contained patients that were treated conservatively. Every study included at least one patient requiring surgical intervention. All the studies used at least one form of imaging to confirm the diagnosis of urachal remnants, with ultrasound (US) being used most (in 90.6% of studies), followed by CT (40.6%) and VCUG (34.4%).

There was a total of 2,171 patients across all 32 studies which includes 805 male patients (37.1%), 590 female patients (27.2%), and 773 patients (35.6%) of unspecified sex. Many studies did not list specific UA subtypes for patients, however, where specified, urachal cysts were the most common anomaly - found in 19 studies, comprising 239 total patients (11%). A patent urachus and urachal sinus were the next most common anomalies, found in 19 and 12 studies, comprising 112 (5.2%) and 100 (4.6%) patients, respectively. Every study had at least one patient with a surgically managed urachal remnant, making up a total of 1,115 patients who were treated surgically. Though many studies did not list the specific surgical approach, open surgery was the most common (in 643 patients) and made up 57.7% of all surgical cases. Unspecified surgical approaches made up 22.4% of cases, and laparoscopic and robotic-assisted laparoscopic approaches made up 17% and 3% of all surgical cases, respectively. Laparoscopic techniques mentioned in the literature began in 2007, and robotic techniques began in 2013.

When specified, the subtype of the UA was listed in descending order of prevalence for each study within Appendix 1 . Most studies also included a distinction between symptomatic and asymptomatic (incidental finding) UAs, and this is listed in Appendix 1 along with presenting symptoms in descending order of prevalence. Recommendations gathered from each included article are listed in Appendix 1 and recommendations regarding imaging or surgical techniques are listed in Appendix 1

Conversion from conservative to surgical management

Out of the 32 total studies, our review identified five studies in the past two decades that included patients who were initially tried on conservative therapy that failed, leading to surgical intervention. Within these studies, we identified a total of 20 patients that were converted to surgical management with conversion rates ranging from 13% to 44% per study ( 13 , 19 , 21 , 24 , 27 ). The conversion percentage, the reason for the conversion, specific UA involved, and surgical complications are listed for each study in Table 2 . The conservative management tab lists the total number of patients who were initially tried on conservative management and the number of patients who were ultimately converted to surgical correction. In the surgical management tab, the total number of surgical patients listed includes all surgically managed patients in the study, including those who were converted from initial conservative management. Out of these patients converted to surgical treatment, 14 of them had a urachal cyst (70%), 1 patient had a urachal sinus (5%), and 5 patients had unspecified anomalies (25%). The most common reason for conversion was an infection, found in 13 patients (65%). Of the total 20 patients who converted from conservative to surgical management, all were recurrently symptomatic. 5 (25%) initially presented symptomatically and 15 (75%) were unspecified. 2 (10%) were males and 18 (90%) were of unspecified gender.

Table 2 Studies identifying conversion of patients from conservative to surgical management.

Originally, obliteration of the urachus was believed to be a strictly prenatal phenomenon, and thus persistence of any part of the urachus was thought to be pathological. In some reports from the early 1970s, it was recommended to remove all urachal remnants when discovered, even those discovered incidentally ( 3 ). This sentiment continued through the early 2000s, from 2000-2006, the recommended management of urachal anomalies was surgical excision regardless of symptom presentation, citing the potential for later infection or evolution into cancer. Even in 2006, Choi et al. supported surgical resection regardless of symptomatic status citing a lack of spontaneous involution and the potential for infection or evolution into cancer ( 30 ). In the past few decades, the more frequent use of advanced imaging studies (such as US) has increased the number of UAs diagnosed incidentally and provided insight regarding the presentation of these abnormalities, with the majority of UAs lacking any noticeable symptoms and containing a minimal risk of malignancy ( 1 ). With increased imaging data, studies have shown that involution of the urachus may even occur postnatally ( 1 , 2 ). In 1998, Ziegler et al. demonstrated that a group of random, asymptomatic newborns all had a urachal remnant on US which spontaneously involuted on a second US exam in 3-5 months ( 2 ). In 2003, Ueno et al. corroborated spontaneous involutions in children up to 1 year of age, and was the earliest study to recommend conservative management, recommending non-invasive management for both symptomatic and asymptomatic presentations ( 1 ). In 2008 and 2009, the literature showed opposing recommendations for management. Three studies were published, with each giving a different suggestion for management: Yapo et al. continued to support surgical management regardless of symptom presentation (notably, the basis for excision of asymptomatic patients was due to a stated 51% chance of malignancy), Galati et al. supported conservative management for all asymptomatic patients and all symptomatic patients under 6 months of age, and Copp et al. stated an inability to provide recommendations based on inconclusive evidence ( 26 – 28 ).

Starting in 2010, the literature appears to address the inconsistency in management recommendations and sought to investigate the best course of management. While these most recent studies consistently support the use of conservative management for asymptomatic presentation. From this time point, it is debated which patients require surgery based on age and symptom presentation. In 2010, Lipskar et al. offered the first proposed treatment algorithm for distinguishing between surgical and conservative management for symptomatic patients, and based the algorithm on symptom presentation, type of UA, and whether UA resolution was achieved after antibiotic treatment ( 24 ).

Further studies found that the associated pathological findings are typically benign, and the calculated number needed to treat to prevent the risk of adenocarcinoma formation is large, at 5,721 persons ( 5 ). Thus, removal of asymptomatic remnants during the first year of life is likely unnecessary. Most remnants may be observed conservatively, with infected remnants initially receiving antibiotic treatment. If a patient fails conservative management and/or presents with severe symptoms, then surgical management should be considered. Surgical excision by an open approach has typically been the gold standard, but over the last two decades, laparoscopic and robotic approaches have shown to be safe and effective alternatives to open surgery with increased cosmetic advantages ( 9 , 10 , 12 , 14 – 17 , 22 , 35 ). The current landscape of UA management consistently juggles conservative and surgical therapies, with no clear predictive signs or guidance on when patients who fail conservative treatment should be switched to definitive surgical management.

Effectiveness of conservative treatment

In 2008, Galati et al. performed a retrospective chart review and discovered 23 patients with UAs (including 12 urachal cysts, 9 urachal sinuses, and 2 patent urachus) who were all initially managed conservatively with silver nitrate and/or unspecified length of observation ( 27 ). Urachal sinuses were more common in boys (8 out of 9) and urachal cysts were more common in girls (9 out of 12), while patent urachi occurred equally in both sexes. About half of the patients required surgical intervention and they recommended surgery for recurrent symptoms or for failure of involution after 6 months of follow-up. The length of time from the initial diagnosis to involution was not noted. Of the 10 patients who converted to surgical management, 9 had urachal cysts and 1 had a urachal sinus. In 2010, Lipskar et al. performed a retrospective chart review of 15 initially symptomatic patients of unspecified gender ( 24 ). The group was composed of 10 urachal cysts and 5 patent urachal sinuses. 10 patients were initially managed conservatively with infected urachal cysts receiving percutaneous drainage and antibiotics. 3 out of the 10 patients had recurrent infections and were converted to surgical management.

In a retrospective case series performed by Nogueras-Ocana et al. in 2014, 13 patients with UAs were identified, 12 of whom were initially managed conservatively with US imaging every 6 months for the first two years and annually thereafter ( 21 ). Two patients (both male, initially presenting symptomatically, and with urachal cysts) were converted to surgical treatment due to reinfection after an initial course of antibiotics. In patients who achieved spontaneous resolution, the time between diagnosis and resolution ranged from 2 to 24 months. In 2015, Stopak et al. performed a retrospective review of 85 patients with UAs which was followed up by a 68-patient retrospective review by Dethlefs et al. in 2019 to monitor the evolution of treatment within the same health system ( 13 , 19 ). In the initial study, it was recommended that UAs be treated conservatively in the first 6 to 12 months of life, and surgically thereafter. Only 15 patients were managed conservatively, with 2 of them requiring surgical management. In the follow-up study, it was noted that care was better shifted to nonoperative management. 24 patients were managed conservatively, with only 3 patients ultimately requiring surgery.

Though we were able to identify five articles that contained patients who were converted from conservative to surgical management ( 13 , 19 , 21 , 24 , 27 ), the lack of consistency between these studies makes it difficult to ascertain salient features of patient demographics or presentations that are predictive of conversion to surgical therapy. The included studies did not have enough patients convert from conservative to surgical management to determine if there are predictive variables for those necessitating surgery such as: certain urachal remnant subtypes, presenting symptoms, age, or gender. In addition, although there were a few patients with noted symptomatic patent urachi who studies claimed to be treated with conservative management ( 27 ), their degree of patency was unknown, and further investigation into this specific anomaly subtype is required to determine the proper management of these patients. Surgical correction may be needed for treatment if this anomaly is persistently symptomatic (e.g., drainage of urine directly from the umbilicus) does not close spontaneously within the first few months of life.

To our knowledge, there has been no comprehensive review to date on UAs. This review identifies recent shifts in preference towards avoiding surgical management for patients under one year of age, with evidence absent for whether this suggestion in management remains true regardless of UA type, presenting symptoms, gender, premature birth status, or specific age. We are currently unable to identify patterns of patients that would be more likely to require a conversion to surgical management. Given the lack of information, we are unable to algorithmically decipher which patients are ideal candidates for surgery and which patients should be observed. Also, due to the lack of randomized controlled trials and prospective studies in the literature, we were also unable to determine an ideal conservative management strategy with included follow-up timelines. Thus it would be necessary to perform a large-scale, multi-site prospective study analyzing demographic information, UA subtypes, and symptomatology regarding different protocol strategies to determine the most efficacious treatment strategy for this pathology. Though, this is not feasible due to the relative rarity of complications and potential for conversion to adenocarcinoma ( 1 ).

Conclusions

The studies included in this review support initial conservative management over prophylactic surgical excision of pediatric UAs, even in symptomatic cases. In the past few decades, a conservative approach has been increasingly favored, especially for the treatment of symptomatic remnants in children 6 months to 1 year of age, where spontaneous resolution and involution were likely, even if initially infected. In older patients, conservative treatment with antibiotics in infected cases has been shown to effectively treat symptoms – thereby eliminating the need for surgical intervention except for rare cases presenting with recurrent infections. There remains a question regarding which associated patient demographics, UA subtypes, or presenting symptoms predict the need for eventual surgical intervention. Currently available studies lack large enough sample sizes to draw any apt correlations. Without sufficient data from prospective studies, the authors are unable to provide a worthwhile management algorithm.

Author contributions

YG, DG, KI, PE, and AS made substantial contributions to conception and design. YG, DG and KI performed acquisition and interpretation of data. YG, DG, KI drafted the article. YG, DG, KI, PE and AS revised the paper critically for important intellectual content. YG, DG, KI, PE, and AS approved of the final version to be published. All authors contributed to the article and approved the submitted version.

Acknowledgments

Publication made possible in part by support from the Nemours Grants for Open Access from Library Services.

Conflict of interest

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

Publisher’s note

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

Supplementary material

The Supplementary Material for this article can be found online at: https://www.frontiersin.org/articles/10.3389/fruro.2023.1159439/full#supplementary-material

Abbreviations

UA, urachal anomaly; US, ultrasound; CT, computed tomography; MRI, magnetic resonance imaging; VCUG, voiding cystourethrogram.

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Keywords: pediatrics - children, urachal abnormalities, management, literature review, urachus, infection

Citation: Ghattas YS, Gelikman DG, Ibanez KR, Ellsworth P and Seth A (2023) Current management strategies of urachal anomalies in pediatric patients: A scoping review. Front. Urol. 3:1159439. doi: 10.3389/fruro.2023.1159439

Received: 05 February 2023; Accepted: 02 March 2023; Published: 17 March 2023.

Reviewed by:

Copyright © 2023 Ghattas, Gelikman, Ibanez, Ellsworth and Seth. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY) . The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

*Correspondence: Abhishek Seth, [email protected]

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

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• Published: 01 May 2024

E-scooter-related dental injuries: a two-year retrospective review

• Junaid Rashid 1 ,
• Rajeevan Sritharan 2 ,
• Sophie Wu 1 &
• Kevin McMillan 3  

British Dental Journal ( 2024 ) Cite this article

Metrics details

Introduction In June 2020, the United Kingdom (UK) published guidance on electric scooter (e-scooter) use to ease transport congestion and reduce pollution. This study aims to examine dental injuries sustained during the two years following initiation of the trial.

Methods The research was conducted at a UK, Level 1, supra-regional major trauma centre. All eligible patient records were analysed to identify e-scooter-related dental injuries to the following regions: teeth, periodontium, alveolus, palate, tongue, floor of mouth, frenum, buccal mucosa and lips. To assess significant associations between recorded variables, a Pearson's chi-square test was utilised.

Results Of the 32 patients who experienced a total of 71 dental injuries, 46.5% (n = 33) affected teeth, predominantly upper central incisors (n = 17). ‘Lacerations' (n = 14) and ‘lip' (n = 11) were the most common type and site of soft tissue injuries, respectively. Unprovoked falls by riders accounted for 53.1% (n = 17) of the injuries. There was an overall increase in e-scooter-related dental injuries throughout the two-year period.

Conclusion E-scooters have introduced an additional source of dental trauma. It is imperative health care professionals can also identify signs of head and non-dental injuries when managing such patients. Further studies are warranted allowing for better informed and optimised dental public health interventions.

E-scooters are a new form of transport that can be the cause of hard tissue and soft tissue dental injuries.

E-scooter-related dental injuries are often related to head injuries, non-dental injuries and intoxication.

The rise in e-scooter-related dental injuries over the two-year period underscores the need for government-instigated e-scooter safety precautions.

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Dental Core Trainee, Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Surgery, Queen Elizabeth Hospital Birmingham, Mindelsohn Way, Birmingham, B15 2GW, UK

Junaid Rashid & Sophie Wu

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Rajeevan Sritharan

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Junaid Rashid: data extraction, data analysis, data interpretation, drafting of the article and final approval. Rajeevan Sritharan: data extraction, data analysis, data interpretation, drafting of the article and final approval. Sophie Wu: conception of idea, data analysis, data interpretation, illustrations, drafting of the article and final approval. Kevin McMillan: conception of idea, data interpretation, critical revisions, final approval, and guarantor of the manuscript.

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The authors report no conflicts of interest. Formal ethical approval was not required as this was a retrospective cohort study. Approval was granted by the Clinical Audit and Research Management System which allowed a unique ID to be created for each of the two years of the study. This ID allowed a health informatics request to be completed to identify suitable patients from a database which was retrospectively analysed. The collected data was completely anonymised and non-identifiable, thus not requiring patient permissions.

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Rashid, J., Sritharan, R., Wu, S. et al. E-scooter-related dental injuries: a two-year retrospective review. Br Dent J (2024). https://doi.org/10.1038/s41415-024-7345-4

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Received : 15 September 2023

Revised : 14 December 2023

Accepted : 18 December 2023

Published : 01 May 2024

DOI : https://doi.org/10.1038/s41415-024-7345-4

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