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  • v.1(4); 2015 Nov

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Making literature reviews more ethical: a researcher and health sciences librarian collaborative process

Bejoy thomas.

1 Department of Psychosocial & Rehabilitation Oncology, Tom Baker Cancer Centre, CancerContol Alberta, Alberta Health Services, Calgary, AB, T2S 3CI, Canada

2 Department of Oncology, Cumming School of Medicine, University of Calgary, Calgary, AB, T2N 1N4, Canada

Admasu Tachble

Delshani peiris, rebecca malhi, glenys godlovitch.

3 Alberta Cancer Research Ethics Committee, CancerContol Alberta, Alberta Health Services, Calgary, AB, T2N 1N4, Canada

Yongtao Lin

4 Knowledge Resource Service, Knowledge Management Department, Tom Baker Cancer Centre, University of Calgary, Calgary, AB, T2N 1N4, Canada

Background:

With emphasis on evidence-based medical care, ‘evidence’ is often the result of literature reviews. Hence, the critical question, “are literature reviews comprehensive?”

This study compares the literature generated by a researcher and a health sciences librarian (HSL).

The Research Associate and the HSL conducted a parallel, segregated literature search on ‘patient-centered care’.

The Research Associate identified 215 manuscripts, and the HSL 129 manuscripts. Overlap was only 55 manuscripts. Differences in process and blind spots are discussed.

Conclusion:

To improve the quality of research outcomes, it seems prudent and ethical to have a synergistic collaboration between researchers and HSLs. Given that this is just one case study that has looked into the issue, further research is strongly encouraged.

Lay abstract: Literature reviews are not just compilations of easily accessible research and should not be taken lightly. Literature reviews are critical to decision-making in clinical trials, medical care and directions of health systems. We wanted to see if researchers and health science librarians working in isolation would create a robust and balanced literature review. This case study showed that both approaches have shortcomings. We recommend that researcher and health science librarian must work together for the success of improving medical care.

Research is cumulative in nature, building on prior studies on the topic of interest. Thus, it is important for the researcher to have robust understanding of the extant knowledge. A comprehensive literature review will describe the research concepts, ground critical appraisal of previously published studies and identify gaps or inconsistencies in the knowledge base, which merit further investigation. Reviewing the literature requires several skills, and is usually done in one of two ways. In the first approach, the researcher (or the research team) critically evaluates relevant information and effectively scans the literature for both breadth and depth of information, to the best of their ability [ 1 ]. Consequently, the researcher/team's capacity to locate and access appropriate studies influences the quality of the research reviewed [ 2 ].

The second option is to engage the services of a health sciences librarian (HSL) with in-depth knowledge of various evidence resources and the professional training to conduct comprehensive literature searches. The comparative advantage researchers have by working with a HSL in the research process is well documented [ 3–7 ]. Yet, the librarian often tends to have a subordinate role and the partnership could be summarized as ‘we (the researcher) gave them the key words, they had a few questions for us, and a week later we got the reference list’. Can the literature review process be optimized? In this case report, we compare the results of literature reviews conducted by a researcher and a HSL.

Review teams

Two separate teams were created for the literature search with a principal investigator (PI) taking the role of independent subject expert in each. One team was comprised of a PhD-trained research associate (RA; A Tachble) and the PI (B Thomas). The other team was comprised of the HSL (Master of Library Information Sciences; Y Lin) and the PI. The PI facilitated the process but did not share information between groups. Thus, both teams conducted parallel, segregated literature searches on the same topic.

Literature review procedure

The literature review process was conducted in four steps: determine the topic of interest; define inclusion and exclusion criteria; compile the literature (total hits); and evaluate the literature to determine ‘relevant hits’ by relevance of the identified literature to the topic of interest (e.g., use of token keyword); a predefined context or setting ; the nature of each hit – is it original (clinical) research or a theoretical stance; and if the original research's methodology could facilitate a decision process to clinical practice (i.e., is process A better than, or equivalent or cost effective, among others compared with process B?)

In the first step, we determined the existing background information on the chosen topic – ‘patient-centered care’ – and began a broad-scale search to identify concepts and to create a provisional list of keywords: patient-centered care, family-centered care, cancer, malignant neoplasm, chronic disease(s), tertiary care, rural care and healthcare provider. For step two, we defined the inclusion criteria (articles published during 2000 and onwards, English language and print/electronic media access to full text, among others) and exclusion criteria (non-English language, personal communications, nonelectronic materials, publications dated prior to 2000 and pediatric/pediatric population) for the literature. The PI then provided these to the RA and the librarian.

The literature search (the third step) included searching through electronic research databases, conference proceedings, dissertation abstracts among others to identify published articles, reports and works in progress. Both teams accessed Cochrane, PsycInfo and MEDLINE data sources to locate published studies/literature addressing patient-centered care. In addition to these, the librarian also searched EMBASE, EBSCO Business Source Complete and Cumulative Index to Nursing and Allied Health Literature (CINAHL), ProQuest Dissertations and Thesis and Web of Science. The RA accessed PubMed and Google Scholar as well.

The literature reviews were evaluated and scored using the following metric:

  • Relevance of the identified literature to the topic of patient or family centeredness (10 points);
  • Context or setting: cancer (10 points) or chronic disease(s) (5 points);
  • Nature of manuscript: original research (e.g., clinical practice; 10 points) or theoretical concept (literature review, hypothesis among others)? (5 points);
  • If the study had an evaluation component and/or used a study design to differentiate between study groups (5 points, respectively) in the manuscript.

The RA and the PI completed the shortlisted manuscript scoring for both teams. A cutoff score for relevance was set at 20 points.

Results are displayed in Figure 1 . The RA obtained 1275 total hits with 215 manuscripts meeting the cutoff score for relevance. The librarian obtained 1232 hits, with 184 manuscripts meeting the relevant scoring criteria. Among the relevant manuscripts, there was a general overlap of 55 (16% of final literature capture) manuscripts between the two teams. The total unique literature capture was 344 manuscripts. The librarian's unique contribution to this literature capture (excluding the general overlap) was 129 manuscripts (37.5% of the final literature capture). Similarly, the unique contribution of the RA was 160 manuscripts (46.5%).

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Object name is fso-01-78-g1.jpg

HSL: Health science librarian; PI: Principal investigator.

In this case study, we note two intertwined but critical aspects. First, the literature capture process by a researcher is a function of their training and experience. The hits obtained here are therefore unique to this researcher (A Tachble); other researchers may have higher or lower hit rates. This leads us to the process resulting in this variance. The librarian worked from the key words and used possible subject headings, related keywords and synonyms to refine a search process. Given that librarians are well acquainted with the anatomy of literature, they work from a very algorithmic process of deductive steps – from creating a very large catchment area, and reducing it by incorporating the exclusion criteria, and honing in on the critical mass by utilizing the inclusion criteria, to subject heading or other control vocabularies. This process of using documented search strategies is replicable between most librarians. Even a few researchers possibly follow these steps. Yet, we believe that most researchers will use databases to create citation lists – particularly databases like PubMed and Google Scholar. This is in essence browsing and utilizes snowballing techniques like perusing through the bibliography of identified hits. Interestingly, searching the term ‘Google Scholar’ in PubMed retrieves about 3926 results, as of August 2015. It would also seem that the 768 + 122 unique hits obtained by the researcher is a result of snowballing and being able to review its content as opposed to capturing it algorithmically; in other words, the manuscript may not be accurately represented by keywords and by extension of medical subject headings (MeSH) and other indexed terms.

The second important interrelated aspect we raise is time utilized by the researcher to compile the literature search. In this case study, the researcher took about 4 weeks to compile 1275 hits. Not all researchers have the luxury of an extended period of time to undertake a comprehensive literature review alone [ 8 ]. In contrast, the librarian would normally take 1–2 days to create and run algorithms, scan the results and rerun the algorithms with a few tweaks to improve or fine-tune the results.

We therefore contend that a literature review by a researcher alone may not be comprehensive using methods of browsing and snowballing. This is quite obvious in Figure 1 where 49 manuscripts that were identified by both teams were not included in the researchers’ final list, primarily because the full manuscript/source document could not be identified. Although their training in resource identification and retrieval is indispensable [ 9–11 ], the expectation that the literature search being the HSL's sole responsibility is – in our opinion – flawed. Our case study seems to indicate that in order to accomplish a literature search with due diligence, a deliberate researcher–HSL collaboration is necessary. Perhaps working in isolation from each other, then collating their findings could be a ‘best practice’ that produces a robust and comprehensive knowledge set.

Even though this is a single case study, and further research is strongly encouraged, from an ethics perspective, these results have important implications for clinical and institutional practices. Patients place their trust in clinicians to provide expert advice and care and in healthcare institutions to facilitate the delivery of good clinical practice through sound, well-informed guidelines. Clinical teams and institutions are legally and ethically bound to provide care that is partly knowledge-based, partly policy-based and partly skills-based. Each of these aspects has its relevant standard that is to be matched or surpassed by competent practitioners. The imperatives of evidence-based medicine are to: ensure that clinicians are aware of what would provide their patient populations with the best care and interventions possible, and inform institutional policy-makers about the relevant options in determining policy. Our findings suggest that the quality of the literature review yielded by the researcher–HSL partnership would satisfy these imperatives.

There are systemic implications too. One is with respect to informing development of clinical guidelines. The presentation of incomplete information may tend to generate an underinformed practice guideline that if implemented could result in causing avoidable harms (or perhaps even in misunderstood benefits). Another systemic implication is that by placing reliance on one form of literature review over the other, the direction of future research could become skewed. As the differences between results for the two searches show, literature blind spots arise. An author is not well placed to be able to report his or her own blind spots, but once published in a peer-reviewed forum, the article or report takes on a certain authority and lives a life of its own in future literature reviews. Any one viewpoint can only tell the story from its own perspective even when done with the good intention of generating evidence-based guidance for actual clinical decision-making. With this in mind, the advantage of having a multiple expertise-enriched literature review is obvious.

Conclusion & future perspective

The results of this single case study demonstrate the advantages that researchers could have by not only involving professional librarians but also by becoming active participants in the literature search endeavor. The ethical implication of the absence of this process is large given the blind spots in the current way of doing literature reviews. For quality control purposes, we believe that that journal editors and peer-reviewers should have a checklist or a process in place to ensure that due diligence in the literature review has been done on any new submission.

Executive summary

  • A comprehensive literature review is the cornerstone of any scientific research endeavor.
  • Literature reviews are usually conducted by a researcher/team or by a health sciences librarian (HSL).
  • The literature capture processes used by researchers and HSLs working in isolation from each other – ‘browsing’ versus ‘algorithmic searching’ – are different, and both lead to blind spots.
  • To accomplish due diligence in a literature search – which has ethical and systemic implications – a deliberate researcher–HSL collaboration is necessary.

Financial & competing interests disclosure

The authors have no relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript. This includes employment, consultancies, honoraria, stock ownership or options, expert testimony, grants or patents received or pending, or royalties.

No writing assistance was utilized in the production of this manuscript.

Informed consent disclosure

The authors state that they have obtained verbal and written informed consent from the patient/patients for the inclusion of their medical and treatment history within this case report.

Open access

This work is licensed under the Creative Commons Attribution 4.0 License. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/

Sage Research Methods Community

Ethics and Your Literature Review

By dr. helen kara, author of qualitative research for quantitative researchers and research ethics in the real world: euro-western and indigenous perspectives ..

An earlier version of this article was originally published in ‘Research Matters’, the quarterly newsletter for members of  the UK and Ireland Social Research Association  (SRA). The SRA now has  a blog  with topical peer-reviewed articles by and for researchers. They are also interested in contributions from readers so, if you fancy writing a guest post, you could  give them a try . They even have a ‘secret researcher’  option for posting anonymously  if you have something really controversial to say.

ethics of literature review pdf

How do researchers address ethics in a literature review?

Researchers often use existing literature to set their research in context. ‘Literature’ is the academic term, referring to peer-reviewed scholarly work such as journal articles. Practice-based researchers may also contextualise their research, though more often with policy and project documents, in part because they are openly available. However, these distinctions are not so hard-and-fast these days. Academics increasingly recognise the value of ‘grey literature’, as they call relevant information that has not been through the peer review process. Practice-based researchers can read more and more academic literature, with the growth of open access, and through schemes such as the SRA’s member benefit of access to  around 6,000 social science journals through EBSCO . Also, the definition of ‘literature’ has grown to include written phenomena and artefacts such as ephemera (leaflets, zines, etc), creative writing (novels, poems, and so on), and online writings such as blog posts and tweets.

When I ask people about the ethical issues of working with literature, they tend to look blank. So here are some pointers. First, define what you are using as literature, or background documents, and explain why you have chosen those types of material. This is important now that there is such a range of available literature: as with all decisions about research, you should be making well-informed choices for good reasons. Then make sure you know how well you can search that body of literature. For example, if you are searching online – as many people do these days – you need to understand the scope and limitations of the electronic tools you use. Google Scholar is many people’s go-to website for academic literature, but it doesn’t index everything, and  its search function is far from neutral. The  Directory of Open Access Journals  indexes work from developing countries that does not find its way into Google Scholar. Even more work from developing countries can be found through the  Journals Online project  run by international research development charity  INASP , which currently covers work from Africa, Latin America, the Philippines, Vietnam, Bangladesh, Mongolia, Nepal and Sri Lanka. Even if your work focuses on a single country or locality, you may find relevant literature from far afield. You are not obliged to search everything; you simply need a clear rationale for your search.

Record your literature search strategy.

You should record your search strategy – where you searched, terms you used to search on, dates of searches – so your readers can assess the effectiveness of your approach. Sadly, these days you will also need to check whether material you plan to cite is bona fide, as directories and repositories may still index and hold literature that has been retracted, or is a spoof that may not be instantly recognisable as such. This means researchers need to be on their guard, and make use of services such as  Retraction Watch  where possible.

Many search strategies will yield far more literature than any researcher, or team, can read. There are ethical dimensions to choosing what to focus on. Bias can creep in here: it is important to read literature representing a good spread of views and opinions, not only those you agree with. Then, when you have chosen what to read, it is ethically necessary to read that work carefully. Take the time to understand the arguments being presented and what they are based on. If you skim-read or cherry-pick, you risk misunderstanding the author’s argument, because you won’t understand their reasoning. Also, superficial reading doesn’t enable you to assess the quality of someone else’s work, so you won’t know how much weight to give it within your own research.

Cite sources correctly.

Then of course you need to cite others’ work correctly and not plagiarise or self-plagiarise. Having said that, self-plagiarism isn’t so much of a problem if you plan to self-publish, whether as an online pdf, e-book, or zine. However, if you plan to publish formally, self-plagiarism is unethical as publishers expect to publish original material.

Taking this kind of an ethical approach to working with literature shows respect to authors of the work on which our own work is based. Also, this approach helps to avoid the replication of errors, which in turn helps to raise standards in research.

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  • Research article
  • Open access
  • Published: 30 April 2021

A scoping review of the literature featuring research ethics and research integrity cases

  • Anna Catharina Vieira Armond   ORCID: orcid.org/0000-0002-7121-5354 1 ,
  • Bert Gordijn 2 ,
  • Jonathan Lewis 2 ,
  • Mohammad Hosseini 2 ,
  • János Kristóf Bodnár 1 ,
  • Soren Holm 3 , 4 &
  • Péter Kakuk 5  

BMC Medical Ethics volume  22 , Article number:  50 ( 2021 ) Cite this article

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The areas of Research Ethics (RE) and Research Integrity (RI) are rapidly evolving. Cases of research misconduct, other transgressions related to RE and RI, and forms of ethically questionable behaviors have been frequently published. The objective of this scoping review was to collect RE and RI cases, analyze their main characteristics, and discuss how these cases are represented in the scientific literature.

The search included cases involving a violation of, or misbehavior, poor judgment, or detrimental research practice in relation to a normative framework. A search was conducted in PubMed, Web of Science, SCOPUS, JSTOR, Ovid, and Science Direct in March 2018, without language or date restriction. Data relating to the articles and the cases were extracted from case descriptions.

A total of 14,719 records were identified, and 388 items were included in the qualitative synthesis. The papers contained 500 case descriptions. After applying the eligibility criteria, 238 cases were included in the analysis. In the case analysis, fabrication and falsification were the most frequently tagged violations (44.9%). The non-adherence to pertinent laws and regulations, such as lack of informed consent and REC approval, was the second most frequently tagged violation (15.7%), followed by patient safety issues (11.1%) and plagiarism (6.9%). 80.8% of cases were from the Medical and Health Sciences, 11.5% from the Natural Sciences, 4.3% from Social Sciences, 2.1% from Engineering and Technology, and 1.3% from Humanities. Paper retraction was the most prevalent sanction (45.4%), followed by exclusion from funding applications (35.5%).

Conclusions

Case descriptions found in academic journals are dominated by discussions regarding prominent cases and are mainly published in the news section of journals. Our results show that there is an overrepresentation of biomedical research cases over other scientific fields compared to its proportion in scientific publications. The cases mostly involve fabrication, falsification, and patient safety issues. This finding could have a significant impact on the academic representation of misbehaviors. The predominance of fabrication and falsification cases might diverge the attention of the academic community from relevant but less visible violations, and from recently emerging forms of misbehaviors.

Peer Review reports

There has been an increase in academic interest in research ethics (RE) and research integrity (RI) over the past decade. This is due, among other reasons, to the changing research environment with new and complex technologies, increased pressure to publish, greater competition in grant applications, increased university-industry collaborative programs, and growth in international collaborations [ 1 ]. In addition, part of the academic interest in RE and RI is due to highly publicized cases of misconduct [ 2 ].

There is a growing body of published RE and RI cases, which may contribute to public attitudes regarding both science and scientists [ 3 ]. Different approaches have been used in order to analyze RE and RI cases. Studies focusing on ORI files (Office of Research Integrity) [ 2 ], retracted papers [ 4 ], quantitative surveys [ 5 ], data audits [ 6 ], and media coverage [ 3 ] have been conducted to understand the context, causes, and consequences of these cases.

Analyses of RE and RI cases often influence policies on responsible conduct of research [ 1 ]. Moreover, details about cases facilitate a broader understanding of issues related to RE and RI and can drive interventions to address them. Currently, there are no comprehensive studies that have collected and evaluated the RE and RI cases available in the academic literature. This review has been developed by members of the EnTIRE consortium to generate information on the cases that will be made available on the Embassy of Good Science platform ( www.embassy.science ). Two separate analyses have been conducted. The first analysis uses identified research articles to explore how the literature presents cases of RE and RI, in relation to the year of publication, country, article genre, and violation involved. The second analysis uses the cases extracted from the literature in order to characterize the cases and analyze them concerning the violations involved, sanctions, and field of science.

This scoping review was performed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement and PRISMA Extension for Scoping Reviews (PRISMA-ScR). The full protocol was pre-registered and it is available at https://ec.europa.eu/research/participants/documents/downloadPublic?documentIds=080166e5bde92120&appId=PPGMS .

Eligibility

Articles with non-fictional case(s) involving a violation of, or misbehavior, poor judgment, or detrimental research practice in relation to a normative framework, were included. Cases unrelated to scientific activities, research institutions, academic or industrial research and publication were excluded. Articles that did not contain a substantial description of the case were also excluded.

A normative framework consists of explicit rules, formulated in laws, regulations, codes, and guidelines, as well as implicit rules, which structure local research practices and influence the application of explicitly formulated rules. Therefore, if a case involves a violation of, or misbehavior, poor judgment, or detrimental research practice in relation to a normative framework, then it does so on the basis of explicit and/or implicit rules governing RE and RI practice.

Search strategy

A search was conducted in PubMed, Web of Science, SCOPUS, JSTOR, Ovid, and Science Direct in March 2018, without any language or date restrictions. Two parallel searches were performed with two sets of medical subject heading (MeSH) terms, one for RE and another for RI. The parallel searches generated two sets of data thereby enabling us to analyze and further investigate the overlaps in, differences in, and evolution of, the representation of RE and RI cases in the academic literature. The terms used in the first search were: (("research ethics") AND (violation OR unethical OR misconduct)). The terms used in the parallel search were: (("research integrity") AND (violation OR unethical OR misconduct)). The search strategy’s validity was tested in a pilot search, in which different keyword combinations and search strings were used, and the abstracts of the first hundred hits in each database were read (Additional file 1 ).

After searching the databases with these two search strings, the titles and abstracts of extracted items were read by three contributors independently (ACVA, PK, and KB). Articles that could potentially meet the inclusion criteria were identified. After independent reading, the three contributors compared their results to determine which studies were to be included in the next stage. In case of a disagreement, items were reassessed in order to reach a consensus. Subsequently, qualified items were read in full.

Data extraction

Data extraction processes were divided by three assessors (ACVA, PK and KB). Each list of extracted data generated by one assessor was cross-checked by the other two. In case of any inconsistencies, the case was reassessed to reach a consensus. The following categories were employed to analyze the data of each extracted item (where available): (I) author(s); (II) title; (III) year of publication; (IV) country (according to the first author's affiliation); (V) article genre; (VI) year of the case; (VII) country in which the case took place; (VIII) institution(s) and person(s) involved; (IX) field of science (FOS-OECD classification)[ 7 ]; (X) types of violation (see below); (XI) case description; and (XII) consequences for persons or institutions involved in the case.

Two sets of data were created after the data extraction process. One set was used for the analysis of articles and their representation in the literature, and the other set was created for the analysis of cases. In the set for the analysis of articles, all eligible items, including duplicate cases (cases found in more than one paper, e.g. Hwang case, Baltimore case) were included. The aim was to understand the historical aspects of violations reported in the literature as well as the paper genre in which cases are described and discussed. For this set, the variables of the year of publication (III); country (IV); article genre (V); and types of violation (X) were analyzed.

For the analysis of cases, all duplicated cases and cases that did not contain enough information about particularities to differentiate them from others (e.g. names of the people or institutions involved, country, date) were excluded. In this set, prominent cases (i.e. those found in more than one paper) were listed only once, generating a set containing solely unique cases. These additional exclusion criteria were applied to avoid multiple representations of cases. For the analysis of cases, the variables: (VI) year of the case; (VII) country in which the case took place; (VIII) institution(s) and person(s) involved; (IX) field of science (FOS-OECD classification); (X) types of violation; (XI) case details; and (XII) consequences for persons or institutions involved in the case were considered.

Article genre classification

We used ten categories to capture the differences in genre. We included a case description in a “news” genre if a case was published in the news section of a scientific journal or newspaper. Although we have not developed a search strategy for newspaper articles, some of them (e.g. New York Times) are indexed in scientific databases such as Pubmed. The same method was used to allocate case descriptions to “editorial”, “commentary”, “misconduct notice”, “retraction notice”, “review”, “letter” or “book review”. We applied the “case analysis” genre if a case description included a normative analysis of the case. The “educational” genre was used when a case description was incorporated to illustrate RE and RI guidelines or institutional policies.

Categorization of violations

For the extraction process, we used the articles’ own terminology when describing violations/ethical issues involved in the event (e.g. plagiarism, falsification, ghost authorship, conflict of interest, etc.) to tag each article. In case the terminology was incompatible with the case description, other categories were added to the original terminology for the same case. Subsequently, the resulting list of terms was standardized using the list of major and minor misbehaviors developed by Bouter and colleagues [ 8 ]. This list consists of 60 items classified into four categories: Study design, data collection, reporting, and collaboration issues. (Additional file 2 ).

Systematic search

A total of 11,641 records were identified through the RE search and 3078 in the RI search. The results of the parallel searches were combined and the duplicates removed. The remaining 10,556 records were screened, and at this stage, 9750 items were excluded because they did not fulfill the inclusion criteria. 806 items were selected for full-text reading. Subsequently, 388 articles were included in the qualitative synthesis (Fig.  1 ).

figure 1

Flow diagram

Of the 388 articles, 157 were only identified via the RE search, 87 exclusively via the RI search, and 144 were identified via both search strategies. The eligible articles contained 500 case descriptions, which were used for the analysis of the publications articles analysis. 256 case descriptions discussed the same 50 cases. The Hwang case was the most frequently described case, discussed in 27 articles. Furthermore, the top 10 most described cases were found in 132 articles (Table 1 ).

For the analysis of cases, 206 (41.2% of the case descriptions) duplicates were excluded, and 56 (11.2%) cases were excluded for not providing enough information to distinguish them from other cases, resulting in 238 eligible cases.

Analysis of the articles

The categories used to classify the violations include those that pertain to the different kinds of scientific misconduct (falsification, fabrication, plagiarism), detrimental research practices (authorship issues, duplication, peer-review, errors in experimental design, and mentoring), and “other misconduct” (according to the definitions from the National Academies of Sciences and Medicine, [ 1 ]). Each case could involve more than one type of violation. The majority of cases presented more than one violation or ethical issue, with a mean of 1.56 violations per case. Figure  2 presents the frequency of each violation tagged to the articles. Falsification and fabrication were the most frequently tagged violations. The violations accounted respectively for 29.1% and 30.0% of the number of taggings (n = 780), and they were involved in 46.8% and 45.4% of the articles (n = 500 case descriptions). Problems with informed consent represented 9.1% of the number of taggings and 14% of the articles, followed by patient safety (6.7% and 10.4%) and plagiarism (5.4% and 8.4%). Detrimental research practices, such as authorship issues, duplication, peer-review, errors in experimental design, mentoring, and self-citation were mentioned cumulatively in 7.0% of the articles.

figure 2

Tagged violations from the article analysis

Analysis of the cases

Figure  3 presents the frequency and percentage of each violation found in the cases. Each case could include more than one item from the list. The 238 cases were tagged 305 times, with a mean of 1.28 items per case. Fabrication and falsification were the most frequently tagged violations (44.9%), involved in 57.7% of the cases (n = 238). The non-adherence to pertinent laws and regulations, such as lack of informed consent and REC approval, was the second most frequently tagged violation (15.7%) and involved in 20.2% of the cases. Patient safety issues were the third most frequently tagged violations (11.1%), involved in 14.3% of the cases, followed by plagiarism (6.9% and 8.8%). The list of major and minor misbehaviors [ 8 ] classifies the items into study design, data collection, reporting, and collaboration issues. Our results show that 56.0% of the tagged violations involved issues in reporting, 16.4% in data collection, 15.1% involved collaboration issues, and 12.5% in the study design. The items in the original list that were not listed in the results were not involved in any case collected.

figure 3

Major and minor misbehavior items from the analysis of cases

Article genre

The articles were mostly classified into “news” (33.0%), followed by “case analysis” (20.9%), “editorial” (12.1%), “commentary” (10.8%), “misconduct notice” (10.3%), “retraction notice” (6.4%), “letter” (3.6%), “educational paper” (1.3%), “review” (1%), and “book review” (0.3%) (Fig.  4 ). The articles classified into “news” and “case analysis” included predominantly prominent cases. Items classified into “news” often explored all the investigation findings step by step for the associated cases as the case progressed through investigations, and this might explain its high prevalence. The case analyses included mainly normative assessments of prominent cases. The misconduct and retraction notices included the largest number of unique cases, although a relatively large portion of the retraction and misconduct records could not be included because of insufficient case details. The articles classified into “editorial”, “commentary” and “letter” also included unique cases.

figure 4

Article genre of included articles

Article analysis

The dates of the eligible articles range from 1983 to 2018 with notable peaks between 1990 and 1996, most probably associated with the Gallo [ 9 ] and Imanishi-Kari cases [ 10 ], and around 2005 with the Hwang [ 11 ], Wakefield [ 12 ], and CNEP trial cases [ 13 ] (Fig.  5 ). The trend line shows an increase in the number of articles over the years.

figure 5

Frequency of articles according to the year of publication

Case analysis

The dates of included cases range from 1798 to 2016. Two cases occurred before 1910, one in 1798 and the other in 1845. Figure  6 shows the number of cases per year from 1910. An increase in the curve started in the early 1980s, reaching the highest frequency in 2004 with 13 cases.

figure 6

Frequency of cases per year

Geographical distribution

The first analysis concerned the authors’ affiliation and the corresponding author’s address. Where the article contained more than one country in the affiliation list, only the first author’s location was considered. Eighty-one articles were excluded because the authors’ affiliations were not available, and 307 articles were included in the analysis. The articles originated from 26 different countries (Additional file 3 ). Most of the articles emanated from the USA and the UK (61.9% and 14.3% of articles, respectively), followed by Canada (4.9%), Australia (3.3%), China (1.6%), Japan (1.6%), Korea (1.3%), and New Zealand (1.3%). Some of the most discussed cases occurred in the USA; the Imanishi-Kari, Gallo, and Schön cases [ 9 , 10 ]. Intensely discussed cases are also associated with Canada (Fisher/Poisson and Olivieri cases), the UK (Wakefield and CNEP trial cases), South Korea (Hwang case), and Japan (RIKEN case) [ 12 , 14 ]. In terms of percentages, North America and Europe stand out in the number of articles (Fig.  7 ).

figure 7

Percentage of articles and cases by continent

The case analysis involved the location where the case took place, taking into account the institutions involved in the case. For cases involving more than one country, all the countries were considered. Three cases were excluded from the analysis due to insufficient information. In the case analysis, 40 countries were involved in 235 different cases (Additional file 4 ). Our findings show that most of the reported cases occurred in the USA and the United Kingdom (59.6% and 9.8% of cases, respectively). In addition, a number of cases occurred in Canada (6.0%), Japan (5.5%), China (2.1%), and Germany (2.1%). In terms of percentages, North America and Europe stand out in the number of cases (Fig.  7 ). To enable comparison, we have additionally collected the number of published documents according to country distribution, available on SCImago Journal & Country Rank [ 16 ]. The numbers correspond to the documents published from 1996 to 2019. The USA occupies the first place in the number of documents, with 21.9%, followed by China (11.1%), UK (6.3%), Germany (5.5%), and Japan (4.9%).

Field of science

The cases were classified according to the field of science. Four cases (1.7%) could not be classified due to insufficient information. Where information was available, 80.8% of cases were from the Medical and Health Sciences, 11.5% from the Natural Sciences, 4.3% from Social Sciences, 2.1% from Engineering and Technology, and 1.3% from Humanities (Fig.  8 ). Additionally, we have retrieved the number of published documents according to scientific field distribution, available on SCImago [ 16 ]. Of the total number of scientific publications, 41.5% are related to natural sciences, 22% to engineering, 25.1% to health and medical sciences, 7.8% to social sciences, 1.9% to agricultural sciences, and 1.7% to the humanities.

figure 8

Field of science from the analysis of cases

This variable aimed to collect information on possible consequences and sanctions imposed by funding agencies, scientific journals and/or institutions. 97 cases could not be classified due to insufficient information. 141 cases were included. Each case could potentially include more than one outcome. Most of cases (45.4%) involved paper retraction, followed by exclusion from funding applications (35.5%). (Table 2 ).

RE and RI cases have been increasingly discussed publicly, affecting public attitudes towards scientists and raising awareness about ethical issues, violations, and their wider consequences [ 5 ]. Different approaches have been applied in order to quantify and address research misbehaviors [ 5 , 17 , 18 , 19 ]. However, most cases are investigated confidentially and the findings remain undisclosed even after the investigation [ 19 , 20 ]. Therefore, the study aimed to collect the RE and RI cases available in the scientific literature, understand how the cases are discussed, and identify the potential of case descriptions to raise awareness on RE and RI.

We collected and analyzed 500 detailed case descriptions from 388 articles and our results show that they mostly relate to extensively discussed and notorious cases. Approximately half of all included cases was mentioned in at least two different articles, and the top ten most commonly mentioned cases were discussed in 132 articles.

The prominence of certain cases in the literature, based on the number of duplicated cases we found (e.g. Hwang case), can be explained by the type of article in which cases are discussed and the type of violation involved in the case. In the article genre analysis, 33% of the cases were described in the news section of scientific publications. Our findings show that almost all article genres discuss those cases that are new and in vogue. Once the case appears in the public domain, it is intensely discussed in the media and by scientists, and some prominent cases have been discussed for more than 20 years (Table 1 ). Misconduct and retraction notices were exceptions in the article genre analysis, as they presented mostly unique cases. The misconduct notices were mainly found on the NIH repository, which is indexed in the searched databases. Some federal funding agencies like NIH usually publicize investigation findings associated with the research they fund. The results derived from the NIH repository also explains the large proportion of articles from the US (61.9%). However, in some cases, only a few details are provided about the case. For cases that have not received federal funding and have not been reported to federal authorities, the investigation is conducted by local institutions. In such instances, the reporting of findings depends on each institution’s policy and willingness to disclose information [ 21 ]. The other exception involves retraction notices. Despite the existence of ethical guidelines [ 22 ], there is no uniform and a common approach to how a journal should report a retraction. The Retraction Watch website suggests two lists of information that should be included in a retraction notice to satisfy the minimum and optimum requirements [ 22 , 23 ]. As well as disclosing the reason for the retraction and information regarding the retraction process, optimal notices should include: (I) the date when the journal was first alerted to potential problems; (II) details regarding institutional investigations and associated outcomes; (III) the effects on other papers published by the same authors; (IV) statements about more recent replications only if and when these have been validated by a third party; (V) details regarding the journal’s sanctions; and (VI) details regarding any lawsuits that have been filed regarding the case. The lack of transparency and information in retraction notices was also noted in studies that collected and evaluated retractions [ 24 ]. According to Resnik and Dinse [ 25 ], retractions notices related to cases of misconduct tend to avoid naming the specific violation involved in the case. This study found that only 32.8% of the notices identify the actual problem, such as fabrication, falsification, and plagiarism, and 58.8% reported the case as replication failure, loss of data, or error. Potential explanations for euphemisms and vague claims in retraction notices authored by editors could pertain to the possibility of legal actions from the authors, honest or self-reported errors, and lack of resources to conduct thorough investigations. In addition, the lack of transparency can also be explained by the conflicts of interests of the article’s author(s), since the notices are often written by the authors of the retracted article.

The analysis of violations/ethical issues shows the dominance of fabrication and falsification cases and explains the high prevalence of prominent cases. Non-adherence to laws and regulations (REC approval, informed consent, and data protection) was the second most prevalent issue, followed by patient safety, plagiarism, and conflicts of interest. The prevalence of the five most tagged violations in the case analysis was higher than the prevalence found in the analysis of articles that involved the same violations. The only exceptions are fabrication and falsification cases, which represented 45% of the tagged violations in the analysis of cases, and 59.1% in the article analysis. This disproportion shows a predilection for the publication of discussions related to fabrication and falsification when compared to other serious violations. Complex cases involving these types of violations make good headlines and this follows a custom pattern of writing about cases that catch the public and media’s attention [ 26 ]. The way cases of RE and RI violations are explored in the literature gives a sense that only a few scientists are “the bad apples” and they are usually discovered, investigated, and sanctioned accordingly. This implies that the integrity of science, in general, remains relatively untouched by these violations. However, studies on misconduct determinants show that scientific misconduct is a systemic problem, which involves not only individual factors, but structural and institutional factors as well, and that a combined effort is necessary to change this scenario [ 27 , 28 ].

Analysis of cases

A notable increase in RE and RI cases occurred in the 1990s, with a gradual increase until approximately 2006. This result is in agreement with studies that evaluated paper retractions [ 24 , 29 ]. Although our study did not focus only on retractions, the trend is similar. This increase in cases should not be attributed only to the increase in the number of publications, since studies that evaluated retractions show that the percentage of retraction due to fraud has increased almost ten times since 1975, compared to the total number of articles. Our results also show a gradual reduction in the number of cases from 2011 and a greater drop in 2015. However, this reduction should be considered cautiously because many investigations take years to complete and have their findings disclosed. ORI has shown that from 2001 to 2010 the investigation of their cases took an average of 20.48 months with a maximum investigation time of more than 9 years [ 24 ].

The countries from which most cases were reported were the USA (59.6%), the UK (9.8%), Canada (6.0%), Japan (5.5%), and China (2.1%). When analyzed by continent, the highest percentage of cases took place in North America, followed by Europe, Asia, Oceania, Latin America, and Africa. The predominance of cases from the USA is predictable, since the country publishes more scientific articles than any other country, with 21.8% of the total documents, according to SCImago [ 16 ]. However, the same interpretation does not apply to China, which occupies the second position in the ranking, with 11.2%. These differences in the geographical distribution were also found in a study that collected published research on research integrity [ 30 ]. The results found by Aubert Bonn and Pinxten (2019) show that studies in the United States accounted for more than half of the sample collected, and although China is one of the leaders in scientific publications, it represented only 0.7% of the sample. Our findings can also be explained by the search strategy that included only keywords in English. Since the majority of RE and RI cases are investigated and have their findings locally disclosed, the employment of English keywords and terms in the search strategy is a limitation. Moreover, our findings do not allow us to draw inferences regarding the incidence or prevalence of misconduct around the world. Instead, it shows where there is a culture of publicly disclosing information and openly discussing RE and RI cases in English documents.

Scientific field analysis

The results show that 80.8% of reported cases occurred in the medical and health sciences whilst only 1.3% occurred in the humanities. This disciplinary difference has also been observed in studies on research integrity climates. A study conducted by Haven and colleagues, [ 28 ] associated seven subscales of research climate with the disciplinary field. The subscales included: (1) Responsible Conduct of Research (RCR) resources, (2) regulatory quality, (3) integrity norms, (4) integrity socialization, (5) supervisor/supervisee relations, (6) (lack of) integrity inhibitors, and (7) expectations. The results, based on the seven subscale scores, show that researchers from the humanities and social sciences have the lowest perception of the RI climate. By contrast, the natural sciences expressed the highest perception of the RI climate, followed by the biomedical sciences. There are also significant differences in the depth and extent of the regulatory environments of different disciplines (e.g. the existence of laws, codes of conduct, policies, relevant ethics committees, or authorities). These findings corroborate our results, as those areas of science most familiar with RI tend to explore the subject further, and, consequently, are more likely to publish case details. Although the volume of published research in each research area also influences the number of cases, the predominance of medical and health sciences cases is not aligned with the trends regarding the volume of published research. According to SCImago Journal & Country Rank [ 16 ], natural sciences occupy the first place in the number of publications (41,5%), followed by the medical and health sciences (25,1%), engineering (22%), social sciences (7,8%), and the humanities (1,7%). Moreover, biomedical journals are overrepresented in the top scientific journals by IF ranking, and these journals usually have clear policies for research misconduct. High-impact journals are more likely to have higher visibility and scrutiny, and consequently, more likely to have been the subject of misconduct investigations. Additionally, the most well-known general medical journals, including NEJM, The Lancet, and the BMJ, employ journalists to write their news sections. Since these journals have the resources to produce extensive news sections, it is, therefore, more likely that medical cases will be discussed.

Violations analysis

In the analysis of violations, the cases were categorized into major and minor misbehaviors. Most cases involved data fabrication and falsification, followed by cases involving non-adherence to laws and regulations, patient safety, plagiarism, and conflicts of interest. When classified by categories, 12.5% of the tagged violations involved issues in the study design, 16.4% in data collection, 56.0% in reporting, and 15.1% involved collaboration issues. Approximately 80% of the tagged violations involved serious research misbehaviors, based on the ranking of research misbehaviors proposed by Bouter and colleagues. However, as demonstrated in a meta-analysis by Fanelli (2009), most self-declared cases involve questionable research practices. In the meta-analysis, 33.7% of scientists admitted questionable research practices, and 72% admitted when asked about the behavior of colleagues. This finding contrasts with an admission rate of 1.97% and 14.12% for cases involving fabrication, falsification, and plagiarism. However, Fanelli’s meta-analysis does not include data about research misbehaviors in its wider sense but focuses on behaviors that bias research results (i.e. fabrication and falsification, intentional non-publication of results, biased methodology, misleading reporting). In our study, the majority of cases involved FFP (66.4%). Overrepresentation of some types of violations, and underrepresentation of others, might lead to misguided efforts, as cases that receive intense publicity eventually influence policies relating to scientific misconduct and RI [ 20 ].

Sanctions analysis

The five most prevalent outcomes were paper retraction, followed by exclusion from funding applications, exclusion from service or position, dismissal and suspension, and paper correction. This result is similar to that found by Redman and Merz [ 31 ], who collected data from misconduct cases provided by the ORI. Moreover, their results show that fabrication and falsification cases are 8.8 times more likely than others to receive funding exclusions. Such cases also received, on average, 0.6 more sanctions per case. Punishments for misconduct remain under discussion, ranging from the criminalization of more serious forms of misconduct [ 32 ] to social punishments, such as those recently introduced by China [ 33 ]. The most common sanction identified by our analysis—paper retraction—is consistent with the most prevalent types of violation, that is, falsification and fabrication.

Publicizing scientific misconduct

The lack of publicly available summaries of misconduct investigations makes it difficult to share experiences and evaluate the effectiveness of policies and training programs. Publicizing scientific misconduct can have serious consequences and creates a stigma around those involved in the case. For instance, publicized allegations can damage the reputation of the accused even when they are later exonerated [ 21 ]. Thus, for published cases, it is the responsibility of the authors and editors to determine whether the name(s) of those involved should be disclosed. On the one hand, it is envisaged that disclosing the name(s) of those involved will encourage others in the community to foster good standards. On the other hand, it is suggested that someone who has made a mistake should have the right to a chance to defend his/her reputation. Regardless of whether a person's name is left out or disclosed, case reports have an important educational function and can help guide RE- and RI-related policies [ 34 ]. A recent paper published by Gunsalus [ 35 ] proposes a three-part approach to strengthen transparency in misconduct investigations. The first part consists of a checklist [ 36 ]. The second suggests that an external peer reviewer should be involved in investigative reporting. The third part calls for the publication of the peer reviewer’s findings.

Limitations

One of the possible limitations of our study may be our search strategy. Although we have conducted pilot searches and sensitivity tests to reach the most feasible and precise search strategy, we cannot exclude the possibility of having missed important cases. Furthermore, the use of English keywords was another limitation of our search. Since most investigations are performed locally and published in local repositories, our search only allowed us to access cases from English-speaking countries or discussed in academic publications written in English. Additionally, it is important to note that the published cases are not representative of all instances of misconduct, since most of them are never discovered, and when discovered, not all are fully investigated or have their findings published. It is also important to note that the lack of information from the extracted case descriptions is a limitation that affects the interpretation of our results. In our review, only 25 retraction notices contained sufficient information that allowed us to include them in our analysis in conformance with the inclusion criteria. Although our search strategy was not focused specifically on retraction and misconduct notices, we believe that if sufficiently detailed information was available in such notices, the search strategy would have identified them.

Case descriptions found in academic journals are dominated by discussions regarding prominent cases and are mainly published in the news section of journals. Our results show that there is an overrepresentation of biomedical research cases over other scientific fields when compared with the volume of publications produced by each field. Moreover, published cases mostly involve fabrication, falsification, and patient safety issues. This finding could have a significant impact on the academic representation of ethical issues for RE and RI. The predominance of fabrication and falsification cases might diverge the attention of the academic community from relevant but less visible violations and ethical issues, and recently emerging forms of misbehaviors.

Availability of data and materials

This review has been developed by members of the EnTIRE project in order to generate information on the cases that will be made available on the Embassy of Good Science platform ( www.embassy.science ). The dataset supporting the conclusions of this article is available in the Open Science Framework (OSF) repository in https://osf.io/3xatj/?view_only=313a0477ab554b7489ee52d3046398b9 .

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Acknowledgements

The authors wish to thank the EnTIRE research group. The EnTIRE project (Mapping Normative Frameworks for Ethics and Integrity of Research) aims to create an online platform that makes RE+RI information easily accessible to the research community. The EnTIRE Consortium is composed by VU Medical Center, Amsterdam, gesinn. It Gmbh & Co Kg, KU Leuven, University of Split School of Medicine, Dublin City University, Central European University, University of Oslo, University of Manchester, European Network of Research Ethics Committees.

EnTIRE project (Mapping Normative Frameworks for Ethics and Integrity of Research) has received funding from the European Union’s Horizon 2020 research and innovation program under grant agreement N 741782. The funder had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

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All authors (ACVA, BG, JL, MH, JKB, SH and PK) developed the idea for the article. ACVA, PK, JKB performed the literature search and data analysis, ACVA and PK produced the draft, and all authors critically revised it. All authors have read and approved the manuscript.

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Supplementary Information

Additional file 1.

. Pilot search and search strategy.

Additional file 2

. List of Major and minor misbehavior items (Developed by Bouter LM, Tijdink J, Axelsen N, Martinson BC, ter Riet G. Ranking major and minor research misbehaviors: results from a survey among participants of four World Conferences on Research Integrity. Research integrity and peer review. 2016;1(1):17. https://doi.org/10.1186/s41073-016-0024-5 ).

Additional file 3

. Table containing the number and percentage of countries included in the analysis of articles.

Additional file 4

. Table containing the number and percentage of countries included in the analysis of the cases.

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Current state of ethics literature synthesis: a systematic review of reviews

  • Marcel Mertz 1 , 2 ,
  • Hannes Kahrass 1 &
  • Daniel Strech 1  

BMC Medicine volume  14 , Article number:  152 ( 2016 ) Cite this article

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Modern standards for evidence-based decision making in clinical care and public health still rely solely on eminence-based input when it comes to normative ethical considerations. Manuals for clinical guideline development or health technology assessment (HTA) do not explain how to search, analyze, and synthesize relevant normative information in a systematic and transparent manner. In the scientific literature, however, systematic or semi-systematic reviews of ethics literature already exist, and scholarly debate on their opportunities and limitations has recently bloomed.

A systematic review was performed of all existing systematic or semi-systematic reviews for normative ethics literature on medical topics. The study further assessed how these reviews report on their methods for search, selection, analysis, and synthesis of ethics literature.

We identified 84 reviews published between 1997 and 2015 in 65 different journals and demonstrated an increasing publication rate for this type of review. While most reviews reported on different aspects of search and selection methods, reporting was much less explicit for aspects of analysis and synthesis methods: 31 % did not fulfill any criteria related to the reporting of analysis methods; for example, only 25 % of the reviews reported the ethical approach needed to analyze and synthesize normative information.

Conclusions

While reviews of ethics literature are increasingly published, their reporting quality for analysis and synthesis of normative information should be improved. Guiding questions are: What was the applied ethical approach and technical procedure for identifying and extracting the relevant normative information units? What method and procedure was employed for synthesizing normative information? Experts and stakeholders from bioethics, HTA, guideline development, health care professionals, and patient organizations should work together to further develop this area of evidence-based health care.

Peer Review reports

Decision making in clinical care, public health, biomedical research, and other fields is strongly based on “external” knowledge (e.g., knowledge from clinical trials, health services research, or economic studies). Non-systematic retrieval and appraisal of external information, however, risks several types of bias and therefore diminishes the quality and accountability of decisions. Systematic reviews (SRs) aim to identify and process information from published material in a systematic, transparent, and reproducible manner. Their ultimate goals are to guarantee comprehensiveness and to reduce systematic errors (bias) in the identification and processing of relevant information, and they are therefore conducive to good evidence-based decision making.

Decision making in medicine, research, and health policy often explicitly or implicitly includes normative ethical considerations. For example, should trial participants be granted access to trial drugs after the end of the study? When health professionals and parents disagree about the appropriate course of medical treatment for a child, under what circumstances is the health professional ethically justified in overriding the parents’ wishes? What are ethical arguments for and against sham interventions? Is it allowable to store biological samples and DNA of minors for non-therapeutic research? When is public health surveillance ethical?

Since the rise of scholarly conduct in “applied” ethical analysis in the 1960s and the establishment of institutes for medical ethics, corresponding peer-reviewed journals, conferences, etc., it seems to be unquestioned that normative ethical input in medical and health policy decision making is a professional enterprise that can be more or less appropriate, of high or low quality, etc. However, it is also known that scholars can come to contrasting but equally well-argued conclusions on what is normatively right or wrong, or more or less appropriate [ 1 – 3 ].

Against this background it is surprising that modern standards for evidence-based decision making in clinical care and public health still rely on eminence-based input alone regarding normative ethical information, even though review methodology has been increasingly used in various disciplines and fields.

Scientific communities such as the Cochrane Collaboration, the Campbell Collaboration, and institutions such as the Institute of Medicine (IOM) or the National Institute for Health and Care Excellence (NICE) provide detailed guidance for review methodologies in different fields [ 4 – 6 ]. While these guidelines cover qualitative as well as quantitative research, they do not explicitly mention whether or how current methodological standards apply to normative ethical literature (“normative literature” for short). Similarly, manuals for evidence-based guideline development do not explain how to include ethical issues in a systematic and transparent manner [ 7 ]. Recent methodological debate demonstrated the need of knowledge synthesis methods that are specified for particular types of information [ 8 ]. But here again, normative ethical information was not acknowledged explicitly.

The ethics literature includes empirical and normative studies on morally challenging topics. Normative literature aims to evaluate or prescribe policies, (moral) reasons, and decisions for or against particular (moral) judgements and policies. Most often, this type of literature can also be described as “argument-based” or “reason-based” literature [ 9 , 10 ]. The “source material” of ethics research includes (ethical) theory, intuitions, common sense, and scientifically produced empirical data.

Despite the neglect of reviews on normative literature by manuals for the development of clinical guidelines and health technology assessment (HTA), and despite any explicit guidance on methodological particularities, such reviews of normative literature already exist, and scholarly debate on their opportunities and limitations has recently bloomed [ 10 – 13 ].

This study aimed to identify trends in the quantity of published systematic and semi-systematic reviews of normative ethical or “mixed” (empirical and normative ethical) literature, the academic affiliations of corresponding authors, and other review characteristics. The study further particularly assessed how these reviews report on their methods for (1) search, (2) selection, (3) analysis, and (4) synthesis of ethics literature.

The review was based on two PubMed searches (15 April 2015, 27 April 2015), with additional searches in PhilPapers (29 April 2015) and Google Scholar (30 April 2015). For PubMed , two search strings were used. The first one was composed for screening purposes, and the second one used a refined search string. See Table  1 and the flowchart in Fig.  1 .

Preferred reporting items for systematic reviews and meta-analyses (PRISMA) flowchart

It proved to be impossible to search directly and solely for reviews of normative literature, as such a distinction is not established or standardized yet in databases (e.g., no standardized key words refer to this kind of review). Therefore, the search had to be intentionally broad in order to capture any review done related to topics of medical ethics or bioethics, even if this included reviews that solely analyzed and synthesized empirical literature.

We have not used a language restriction for the search in order to assess the overall amount of identifiable reviews.

For the purpose of this meta-review on a still little-standardized review area we decided to apply rather sensitive and not too restrictive selection criteria. We selected all reviews that explicitly or implicitly indicated their objective to analyze and present ethics literature in a systematic manner. To be included, reviews had to be explicitly concerned with normative ethical considerations of medical topics; e.g., they had to pose an ethical question or determine ethical challenges. It was not deemed sufficient for the results of a review to be able to be regarded as “ethically relevant.” Furthermore, reviews should have an identifiable description of at least some methodological elements describing a reproducible literature search (e.g., search terms, databases used, or inclusion/exclusion criteria). See Table  2 . We labeled such reviews as semi-systematic reviews . Only those reviews that explicitly or implicitly reported on search, selection, analysis, and synthesis were labeled as (full) systematic reviews . Finally, we only included reviews written in English, German, or French.

Articles were selected first according to their title or abstract, and later by full text screening. See Table  2 . All reviews for empirical, normative, and “mixed” literature were included at this stage. The in-depth analysis and corresponding data presented in this paper focused on the normative and mixed literature, because methodological particularities, especially concerning analysis and synthesis, have been much less widely discussed for normative and conceptual literature than for empirical research.

The selection was initially done by one researcher (MM). Then, a second researcher (HK) checked all the selection results (inclusion and exclusion) for consistency with the selection criteria. Discrepancies were discussed and successfully overcome via consensus-seeking discussions.

Because we aimed to assess the current state of the art of reviews of normative ethical literature, we did not exclude reviews that did not fulfill all PRISMA criteria. Depicting the state of art must also include reviews of “relatively bad” reporting quality. Also, it is possible that certain reviews demonstrate a fair reporting of analysis and synthesis of normative information but are not able to fulfill some basic PRISMA criteria. Excluding such reviews would deprive our review of important insights about how reviews of normative information are analyzing and synthesizing information. Nevertheless, we present slightly adapted PRISMA ratings as part of our results.

Apart from the reporting quality, it would also be impossible to assess the methodological quality of the included reviews because of the lack of specific quality assessment tools for reviews of normative ethics literature.

We determined the academic fields of the journals that published included reviews based on how they were classified by the Journal Citation Reports ( JCR ) Science Edition 2014 and JCR Social Science Edition 2014 . Where no entry was available, the journal was categorized as “not found”.

We further categorized the affiliation of all authors. (Table  4 lists the different categories used.) For this purpose, we considered the affiliation of all first authors. We took the lowest identifiable organizational unit if several organizational units/levels were mentioned. If the last author had a differing affiliation, this affiliation was also considered. Finally, if additional authors of a review had further differing affiliations, these were also considered. Therefore, the amount of authors considered regarding affiliations is not equal to the total amount of authors.

The method of qualitative content analysis (QCA) [ 14 , 15 ] was employed to analyze the literature in detail, i.e., to identify and categorize the methods used for search, selection, analysis, and synthesis, and the information given about methodology (e.g., stating aims, discussing limitations, providing a flowchart). In applying this method, we used a combined deductive and inductive strategy for building up categories [ 14 ]. This was done iteratively by two researchers (MM, HK).

The qualitatively analyzed content of the reviews was synthesized into descriptive statistics assessing how often the description of methods corresponded to established (and slightly adapted) criteria of the PRISMA guideline [ 16 ] (See Table  6 ).

From the initially identified 1393 references we finally included 160 reviews covering three types of ethics reviews: (1) empirical ethics ( n  = 76), (2) normative ethics ( n  = 51), and (3) mixed literature ( n  = 33). For the above-described reasons we further excluded the 76 reviews of empirical ethics literature from the in-depth analysis. See the flowchart in Fig.  1 . The following results therefore represent the remaining 84 reviews of normative or mixed literature. Additional file 1 : Tables S1–S3 present all references for the three types of ethics reviews.

Languages, publication dates, and self-labeling

Of all 84 reviews, 98 % ( n  = 82) were in English, one in French, and one in German. The earliest reviews were published in 1997. Of the 84 reviews, 82 % were published in the last ten years. See Fig.  2 . In total, 31 (37 %) labeled themselves as “systematic review” or used the term “systematic” in labelings such as “systematic literature review” or “systematic survey.”

Publication dates of the reviews

Journals: academic fields and titles

The academic fields most prominent were Nursing ( n  = 17, 15 %), Medical Ethics and Ethics ( n  = 10 + 2 = 12, 11 %), Public, Environmental, and Occupational Health ( n  = 8, 7 %), and Genetics and Heredity ( n  = 8, 7 %). See Table  3 . Note that a journal can be classified in two or more fields.

The journal that published the most reviews was Nursing Ethics ( n  = 7, 8 %), followed by Journal of Medical Ethics ( n  = 4, 5 %), BMC Medical Ethics ( n  = 4, 5 %), Journal of Advanced Nursing ( n  = 4, 5 %), and European Journal of Human Genetics ( n  = 4, 5 %). However, roughly 70 % ( n  = 59) of all finally included reviews ( n  = 84) were found in journals that only appeared once in our review. See Table  3 .

Authors: number, country of origin, and affiliations

The greatest number of reviews were authored by two authors ( n  = 26, 31 %), followed by three ( n  = 18, 21 %) and four authors ( n  = 16, 19 %) with an arithmetic mean of 3.45. See Table  4 .

Twenty reviews (24 %) were written by authors from the USA, 10 (12 %) from the UK, 10 (12 %) from Belgium, 8 (10 %) from Germany, and 6 (8 %) from the Netherlands. The remaining 30 reviews were written by authors from 18 other countries. See Table  4 .

We analyzed the affiliation of 205 authors with different affiliations. The greatest number, namely 60 (30 %), were affiliated to Bioethics institutions, 51 (25 %) to institutions related to medicine, 23 (11 %) to Nursing and Allied Health Practitioners (AHP)-related institutions, 18 (9 %) to Health Sciences institutions, and 7 (3 %) were affiliated to Philosophy and the Humanities. See Table  4 .

Standards/guidelines and limitations

Twenty (24 %) of the 84 reviews stated that they used an established/published review methodology (see Table  5 ). Only the approach of McCullough et al. and Garrard were mentioned more than once ( n  = 9, 45 %, n = 2, 10 %). Ten reviews (12 %) stated that they took guidance from established reporting standards or guidelines (whether general or specific to SRs). The only standard mentioned more than once was PRISMA, with 8 entries. Thirty-three reviews (39 %) reported on limitations.

Reported methods for search, selection, analysis, and synthesis

Table  6 presents detailed data on how often the reviews were transparent about methodological criteria for search, selection, analysis, and synthesis. Table  6 also highlights how these criteria match with reporting items mentioned in PRISMA. Most reviews reported, for example, on what databases (93 %), search terms (91 %), or inclusion/exclusion criteria (81 %) they used. Overall, only 1 % and 8 % did not fulfill any criteria related to search and selection, respectively. However, only a minority reported on other essential details such as the procedure for information extraction (37 %) and information synthesis (18 %). In fact, 31 % did not fulfill any criteria related to the reporting of analysis methods. For example, only 25 % of the reviews reported the ethical approach needed to analyze and synthesize normative information.

A comprehensive qualitative analysis and comparison of all applied methods for search, selection, analysis, and synthesis is beyond the scope of this paper and is to be published elsewhere. The applied methods for search and selection of relevant normative literature are largely comparable with standard “systematic review” methodology. Methods for analysis and synthesis of normative information, however, are of substantial differences. In the following, therefore, we highlight some core findings with regard to the reported analysis and synthesis.

Regarding extraction and analysis of normative information, the most sought types of information were ethical issues, topics, or dilemmas ( n  = 27), arguments or reasons ( n  = 14), and ethical principles, values, or norms ( n  = 13) (multiple responses possible). Among the procedures for extracting information we broadly distinguished between “coding and categorizing” ( n  = 9), “collecting” ( n  = 7), or “close reading” ( n  = 6). See Table  7 for more detailed explanations and case examples.

Regarding synthesis, we could broadly distinguish between qualitative methods ( n  = 44), quantitative methods ( n  = 5), and narrative/hermeneutical methods ( n  = 3). In most cases, qualitative analyses aimed to develop overarching normative issues, reasons, or principles that allowed summarizing the more detailed normative information. To do this, a variety of deductively and inductively developed category systems with main and subcategories were employed. Quantitative analyses aimed, for example, to quantify the distribution of qualitatively assessed topics. See Table  8 for more detailed explanations and case examples.

Thirty-eight (45 %) of the included reviews ( n  = 84) reported on at least some aspects of all four domains of the methodology (search, selection, analysis, and synthesis).

Most reviews reported on the essential elements for search and selection methods (e.g., databases, search terms, inclusion/exclusion) except for flowcharts (reported by only 29 %). However, reporting was much less explicit for analysis and synthesis methods. Almost one third of all reviews did not report on any essential element of the analysis methods (what information to extract and how). For example, only 25 % of reviews on normative literature reported on the kind of ethical approach/theory needed to identify relevant normative information. Only 45 % of reviews reported on all methods and could therefore be labeled as (full) systematic reviews, implying that most reviews we found are rather semi-systematic. Somehow in line with the aforementioned neglect of important method reporting is the fact that only 39 % of reviews discussed their limitations.

A limitation of our review is that we only searched the databases PubMed, PhilPapers, and Google Scholar . We restricted our search to these three databases mainly because of experiences from former systematic reviews of normative information demonstrating that most of the literature can be found in PubMed and Google Scholar , and that searching other ethics-specific databases did not add a substantial proportion of references [ 17 ]. In our review, 86 % of all included reviews were found by PubMed searches alone. Furthermore, all languages other than English, German, or French were excluded, but this only resulted in the exclusion of three reviews.

Our results demonstrate that most elements of searching and selecting normative literature reflect the widely accepted PRISMA recommendations. However, appropriate elements for the analysis and synthesis of normative literature are less standardized. Further meta-research and conceptual analysis are needed to inform the development of minimal standards for the analysis and synthesis of normative literature. The quality assessment of normative literature might be one of the most controversial topics in this regard [ 10 ]. The required degree of transparency for all steps of information processing in analyzing and synthesizing normative information will be another controversial topic, because strong requirements in this regard might result in excessive workloads for review authors [ 18 ].

Nevertheless, our review demonstrates that analysis and synthesis methods can be described and justified with regard to the specific review objectives. This demands that the following elements for analysis and synthesis should be clarified prior to each review of normative information and should be reported with the dissemination of results: (1) normative information unit (e.g., ethical issues, ethical reasons, ethical norms, etc.), (2) ethical approach (e.g., a specific ethical theory) and the technical procedure used to identify and extract the relevant normative information units, (3) method for synthesizing normative information (e.g., category building). See Tables  7 and 8 . Researchers should also be aware that these three steps are interrelated; i.e., that using a specific ethical approach will lead to a specific way of identifying normative information units, or, vice versa, that the set of normative information units identified will depend on the ethical approach (e.g., a deontological ethical theory would identify some issues as “ethical issues,” which a consequentialist ethical theory would not).

Thus, future clarification is also needed for the personal competencies and skills necessary to realize a valid and informative review of normative information. Based on our personal experiences with reviews of normative information, it is also important to clarify the expectations and needs of the intended readership. In particular, the choice of synthesis methods for normative information might differ substantially if the review group aims to inform either expert discourse in bioethics or policy decision making in guideline or HTA development. Stakeholder orientation, therefore, is another issue that should be clarified prior to conducting ethics reviews.

This is the first study, to our knowledge, to analyze the state of systematic and semi-systematic reviews of normative literature on medical topics. We identified 84 reviews published between 1997 and 2015 in 65 different journals and demonstrated an increasing publication rate for this type of review. The reference lists for all included reviews (Additional file  1 : Tables S1–S3) provide a rich source for those interested in medical ethics and those wanting to conduct (systematic) reviews of normative literature themselves.

Further research as well as interdisciplinary discussion and consent are needed to define detailed best practice recommendations for the respective steps of a review of normative information. Experts from different fields such as bioethics, HTA and guideline development, as well as health care professionals and patient representatives, should work together to further develop the methodology of (systematic) reviews of normative ethical information to support evidence-based health care.

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Acknowledgements

We would like to thank our student assistant Nadine Komeinda for her help in retrieving and electronically archiving the full text versions of the articles we found, and our student assistant Christopher Schürmann for his help in analyzing review characteristics.

Authors’ contributions

MM wrote the main draft of the paper (all sections), devised search algorithms and conducted the search, worked out most of the methods employed, and revised and finalized the manuscript. HK assisted in devising the search algorithms, cross-checked selection, was one of two researchers analyzing and synthesizing the material, and contributed to writing the manuscript. DS originated the idea of conducting a systematic review about reviews of normative ethical literature on medical topics, gave input to the review design, acted as third (“control”) researcher in the analysis procedure, and revised the manuscript. All authors read and approved the final manuscript.

Competing interests

Financial competing interests: There are none to declare. Non-financial competing interests: In three reviews finally included in this review DS was one of the authors. In one review MM and HK were co-authors.

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Additional file

Additional file 1: table s1..

Reviews (English/German/French): empirical literature. Table S2 : Reviews (English/German/French): normative literature. Table S3 : Reviews (English/German/French): mixed literature. (DOCX 56 kb)

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Mertz, M., Kahrass, H. & Strech, D. Current state of ethics literature synthesis: a systematic review of reviews. BMC Med 14 , 152 (2016). https://doi.org/10.1186/s12916-016-0688-1

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DOI : https://doi.org/10.1186/s12916-016-0688-1

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  • Systematic review
  • Literature review
  • Normative literature
  • Argument-based literature
  • Empirical ethics
  • Literature search
  • Evidence-based medicine

BMC Medicine

ISSN: 1741-7015

ethics of literature review pdf

  • Open access
  • Published: 24 May 2020

A systematic literature review of the ethics of conducting research in the humanitarian setting

  • William Bruno   ORCID: orcid.org/0000-0001-9429-6874 1 &
  • Rohini J. Haar 2  

Conflict and Health volume  14 , Article number:  27 ( 2020 ) Cite this article

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Research around humanitarian crises, aid delivery, and the impact of these crises on health and well-being has expanded dramatically. Ethical issues around these topics have recently received more attention. We conducted a systematic literature review to synthesize the lessons learned regarding the ethics of research in humanitarian crises.

We conducted a systematic review using the Preferred Reporting Items for Systematic Reviews and Meta-analysis (PRISMA) guidelines to identify articles regarding the ethics of research in humanitarian contexts between January 1, 1997 and September 1, 2019. We analyzed the articles to extract key themes and develop an agenda for future research.

We identified 52 articles that matched our inclusion criteria. We categorized the article data into five categories of analysis: 32 were expert statements, 18 were case studies, 11 contained original research, eight were literature reviews and three were book chapters. All included articles were published in English. Using a step-wise qualitative analysis, we identified 10 major themes that encompassed these concepts and points. These major themes were: ethics review process (21 articles, [40.38%]); community engagement (15 articles [28.85%]); the dual imperative , or necessity that research be both academically sound and policy driven, clinical trials in the humanitarian setting (13 articles for each, [25.0%)]; informed consent (10 articles [19.23%]); cultural considerations (6 articles, [11.54%]); risks to researchers (5 articles, [9.62%]); child participation (4 articles [7.69%]); and finally mental health , and data ownership (2 articles for each [3.85%]).

Conclusions

Interest in the ethics of studying humanitarian crises has been dramatically increasing in recent years. While key concepts within all research settings such as beneficence, justice and respect for persons are crucially relevant, there are considerations unique to the humanitarian context. The particular vulnerabilities of conflict-affected populations, the contextual challenges of working in humanitarian settings, and the need for ensuring strong community engagement at all levels make this area of research particularly challenging. Humanitarian crises are prevalent throughout the globe, and studying them with the utmost ethical forethought is critical to maintaining sound research principles and ethical standards.

Defined as both natural and man-made disasters, along with both acute and chronic conflicts, humanitarian crises threaten the lives and livelihoods of over 131 million people in the world today [ 1 ]. With more than 68.5 million people currently displaced, 25.4 million of whom are refugees outside their country of origin, the global community is witnessing urgent humanitarian issues that are crossing borders and impacting even those states and communities once thought immune [ 2 , 3 ]. Humanitarian aid is the impartial, independent and neutral delivery of services to populations in immediate danger [ 4 ]. Since the end of World War II, the humanitarian aid sector (in the form of health services, water and sanitation services, nutritional goods and security) has grown tremendously [ 5 ].

With expansion in humanitarian aid delivery and the deepening awareness that humanitarian crises can destroy health systems and have long-term impacts on public health, ensuring that the services provided are effective and acceptable is crucial. Following several highly publicized failures of the humanitarian community, veteran humanitarians from across the spectrum of governmental and non-governmental organizations have attempted to improve humanitarian response [ 6 ]. Initiatives such as the Sphere Project and others aimed to create minimum standards and evidence-based protocols for the delivery of five core components of humanitarian response—water supply and sanitation, nutrition, food aid, shelter and site planning and health services [ 7 ]. Over the past several decades, a key component of the assessment process has been conducting formal monitoring, evaluation and research on humanitarian aid delivery. Studies ranging from randomized control trials to population surveys and qualitative assessments evaluating the full spectrum of humanitarian aid delivery have burgeoned [ 8 ].

Parallel to the increase in professionalization of humanitarian aid, the public health community has been grappling with how to ensure that research on vulnerable populations is conducted ethically and with a focus on the rights and best interests of the community. Spurred by a backlash to unchecked human experimentation carried out through the twentieth century during World War II and the decades afterwards, there is more recognition of the critical importance of considering research ethics, particularly when studying vulnerable populations [ 9 ].

Few populations are as vulnerable to the potential adverse ethical challenges of research as those experiencing a humanitarian crisis [ 10 ]. Faced with weak government protections, disrupted health systems, insecure living conditions, and unreliable food and unsafe water, disaster-affected populations can be particularly at risk of inadequate consent processes and coercion. Furthermore, humanitarian emergencies require timely evaluation and management, making traditional ethics review—typically a protracted process—impractical [ 11 , 12 , 13 ]. These unique challenges, along with underdeveloped oversight and regulatory bodies of host countries and international mechanisms, make ethics considerations a crucial but difficult task in humanitarian research [ 14 , 15 ].

Despite increasing interest and an expanding literature base, there has been limited formal synthesis of the existing published data around the ethical issues of research in the humanitarian setting. We conducted a systematic review to (1) identify ethical issues surrounding research in humanitarian settings, (2) assess how these issues are managed in these unique circumstances and (3) develop an agenda for major issues that will require further discourse.

We conducted a systematic review using the Preferred Reporting Items for Systematic Reviews and Meta-analysis (PRISMA) guidelines [ 16 ]. The PRISMA checklist has been provided as Supplementary Table  1 . Articles relevant to research ethics in the humanitarian setting were identified and analyzed. We chose to limit the search to articles published after January 1, 1997, when the initiation of the Sphere project marked a paradigm shift in how humanitarian aid was envisioned and carried out. This allows for review of nearly 25 years of literature, therefore spanning a wide swath of potential ethical research. We used the Sphere project dates because it included explicit language highlighting the need for evidence-based practices, which would require significant augmentation in research efforts to provide such an evidence base [ 7 ]. Our search included articles published as late as September 1, 2019, when this study was first undertaken.

Search strategy

We searched PubMed and Scopus for articles with significant discussion of the ethical issues of humanitarian research ethics. After a qualitative assessment of relevant keywords, we identified all pertinent articles based on the following terminology categories (articles could be in any language): (1) humanitarian settings (terms such as humanitarian, global health, disaster, emergency and/or conflict), (2) ethics (terms such as ethic(s), bioethics, human rights and/or rights) and (3) research type (terms such as research, program evaluation, monitoring and evaluation and/or investigation). The full search strategy and MeSH terms can be found in the Appendix . The initial search results of 1459 articles underwent a title and abstract review followed by a full text review by two different authors (WB and RH) (Fig.  1 ). A priori inclusion criteria included the 22-year timeframe mentioned above and selected for articles with robust discussion of ethical issues in the context of conducting research in humanitarian settings. Any article deemed by both reviewers to contain only a superficial mention of ethical issues and to not substantively (1) discuss ethics or (2) focus on research (3) in the context of humanitarian settings was excluded from the final analysis. Ethics was defined broadly as engagement with specific research ethics, as well as human rights issues, and other non-formal discussions of right versus wrong and other moral concepts. Research was defined as discussions including any types of data collection including quantitative and qualitative, as well as data collection for monitoring and evaluation for other programmatic and academic purposes. Humanitarian settings included diverse contexts including conflict and post-conflict states, post-natural disaster settings and refugee camps that requires specific interventions to prevent large scale suffering of the populations. Two authors (WB and RH) reviewed the final list of articles meeting the inclusion criteria.

figure 1

Stages of Systematic Literature Review Utilizing PRISMA Guidelines

Analytical methods

We used a modified meta-ethnographic approach to inductively identify key concepts and synthesize the major themes [ 17 ]. We chose the meta-ethnographic approach as it has been shown useful in other systematic reviews of qualitative health literature in that it utilizes an inductive approach that can account for differences in methodology and focus, and has the potential to provide a higher level of analysis and generate new research questions [ 18 , 19 , 20 ]. We conducted three steps of analysis: (1) Identifying original concepts and ideas from each paper that related to cross-cutting themes; (2) synthesizing these ideas into cross-cutting themes; and (3) identifying major themes. These steps are outlined in Table  2 . Original concepts were topics discussed in each paper, which the authors felt had some relevance to this paper’s focus on humanitarian research ethics. Cross-cutting themes were key concepts that were identified in at least two different articles. We assessed how the cross-cutting themes may fall into broader overarching ideas and coded these into related non-mutually exclusive groups we termed major themes. The synthesis process of extracting these major themes was one of reciprocal translation and constant comparison of concepts across studies. The process elucidated tensions and areas for future research within each major theme, as shown in Table 2 . Any disagreements on the analysis were resolved with discussion and consensus.

This research, based on previously published literature, did not meet criteria for Institutional Review Board approval.

Of the 1459 unique articles resulting from our search terms, 52 matched our inclusion criteria (Table 1 : List of Included Articles). The articles took the shape of five non-mutually exclusive categories of analysis: 32 were expert statements, 18 were case studies, 11 contained original research, eight were literature reviews and three were book chapters. All included articles were published in English. Thirty-four of the 52 (65.38%) articles were published in 2015 or later, ten between 2007 and 2014, and eight were published in the 1997–2006 decade (Fig.  2 ). Of the 52 articles included for final analysis, 23 were published by international teams (meaning that they were comprised of members from at least two different countries), 12 were from the United States, six from the United Kingdom, three from Canada, two each form Ireland, Trinidad and Tobago, and Switzerland, and one each from Australia and India.

figure 2

Included articles by publication date

Thematic analysis

The step-wise analysis is presented in Table 2 . First order analysis of the articles meeting our final inclusion criteria revealed ideas and issues within the context of ethics related research in humanitarian settings. In the second phase of the analysis, qualitative review of the reports identified cross-cutting themes between the papers, and 10 major themes that encompassed these concepts and points. These major themes in descending order of prevalence were ethics review process (21 articles, [40.38%]); community engagement (15 articles [28.85%]); the dual imperative , or necessity that research be both academically sound and policy driven and clinical trials in the humanitarian setting (13 articles for each, [25.0%]); informed consent (10 articles [19.23%]); cultural considerations (6 articles, [11.54%]); risks to researchers (5 articles, [9.62%]); child participation (4 articles [7.69%]), and finally mental health , and data ownership (2 articles for each [3.85%]).

Ethical review

Discussion of the ethical review process was the most commonly identified theme, with 21 articles having a substantive focus on this [ 11 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 ]. Independent ethics review prior to the start of a study is a core component of research ethics. Tansey et al. conducted a survey of ethics review board members with experience in reviewing research ethics in disaster settings. Their results suggest a general feeling that research in this setting is not only of particularly high social value, making it a desirable pursuit, but also necessitates a higher level of justification due to the inherent vulnerability of the research subjects [ 33 ]. There is also general agreement that the innate fluidity and urgency of humanitarian situations make swift and efficient ethics review of paramount importance [ 11 , 25 , 29 ]. Hunt et al. report, “where research is launched in response to a sudden-onset disaster such as an earthquake or hurricane, researchers may need to initiate their protocols quickly in order to answer research questions pertinent to the acute phase of the disaster response” [ 11 ]. However, as mentioned above, the particular vulnerability of the subjects being studied leads many research ethics committees to automatically identify humanitarian research as requiring “the highest level of stringency”. On the other hand, framing research as “needs assessments” and/or “monitoring and evaluation,” which is often done in evaluating aid needs and programs, may act to sideline rigorous ethical review and jeopardize the well-being of the recipient population [ 11 ]. This contradiction of values makes ethical review of humanitarian research particularly challenging.

Authors suggested strategies to mitigate the inherent challenges of ethics review in this setting [ 25 ]. For example, Hunt et al. suggest pre-approved research protocol templates which can be quickly customized for use in individual emergencies [ 11 ]. Eckenwiler et al. propose what they refer to as ‘real-time responsiveness,’ which is an iterative strategy of constant dialogue between ethics reviewers and researchers while studies are being conducted [ 24 ]. Given the potential for misstep in an expedited initial ethics review, Chiumento et al. describe the utility of a post-research ethical audit. The authors explain how this could help to evaluate “procedural ethics against in-practice realities”, which could help inform future studies [ 21 ]. Ethical analysis after data collection may also offer the added benefit of offering lessons on the review and practice process to the reviewers and researchers.

Our results highlighted the particular case of how the humanitarian aid agency Médecins Sans Frontières’ (MSF), who conducts substantial research in humanitarian settings, has devised an independent Ethics Review Board (ERB). The ERB utilizes several of the strategies mentioned above such as pre-approved protocols, engaging in ongoing dialogue between researchers and the ERB and conducting post-research evaluations [ 29 , 31 ]. Saxena et al. reported on a joint panel conducted by the WHO and the African Coalition for Epidemic Research, Response and Training. The authors outline the group’s recommendations for “rapid and sound ethics review”, which includes “preparing national ethics committees for outbreak response; pre-crisis review of potential protocols; multi-country review; coordination between national ethics committees and other key stakeholders; data and benefit sharing; and export of samples to third countries” [ 32 ]. Indeed, as Mezinska et al. point out in their systematic review of ethical guidelines, most of the analyzed documents included in their report did “not attempt to give researchers and other stakeholders a comprehensive overview of how to proceed ethically in all types of research and in all types of disasters”, which the authors see as problematic given that “disaster research is unavoidably context and time sensitive, making generalized guidance less applicable” [ 35 ].

Community engagement

Substantive involvement of the community being studied was identified as an imperative for researchers and a major theme of discussion in 15 articles [ 21 , 22 , 30 , 32 , 33 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 ]. It was generally agreed that active participation is necessary in order to fulfill the ethical requisite that research be of use to the community being studied (also known as beneficence) [ 22 , 48 , 50 ]. As Chiumento et al. identified in their systematic review of mental health literature, the right to participate in research can be viewed as a basic right in and of itself, insofar as it relates to other rights such as self-determination and autonomy [ 22 ]. One important strategy described was involving local community health and government officials in an effort to maximize community support [ 43 ]. More practically speaking, this effort can help limit potential for a community’s misunderstanding of research, which can jeopardize a project’s legitimacy and undermine its acceptance [ 46 ]. Early involvement of community actors, potentially via consultation during study protocol design or community meetings, was suggested [ 21 , 42 ].

The discussions within the articles suggest that community involvement also involves strengthening local institutions, effectively improving their ability to conduct their own research [ 21 , 22 ]. Despite being recognized as an important component of ethical research, it was generally agreed that there is a critical shortage of local capacity to carry out studies, particularly in post-conflict zones where formal institutions are often eroded [ 45 , 47 ]. In their study on the research capacity of Somaliland, Boyce et al. identified potential harms of a “dominance of authors from [High-Income Countries]” [ 45 ]. They explain that, for example, the unrelatability between researcher and subject could lead to a reduced relevance of the research question.

Despite the agreement for “a set of practices that help researchers establish and maintain relationships with the stakeholders to a research program”, Tansey et al. discuss some of the inherent challenges in community participation. Particularly when conducting disaster research, the practicality of including locals can be difficult when “you don’t know when the disaster is going to hit. .. so it would be hard to set up community approvals and engagement beforehand” [ 33 ]. Furthermore, lack of adequately trained researchers and poor local infrastructure are perennial problems [ 45 ]. While ethically desirable, partnering with the local community may, in many circumstances, often prove practically prohibitive.

While including local authorities in research may seem prudent on face value, as discussed in the section on cultural considerations, these articles make clear the potential for ethical ambiguity when dealing with such actors [ 47 , 49 ]. For example, in a civil war context, researchers may hope to adhere to humanitarian principles of impartiality to ensure access to participants and safety for researchers [ 49 ]. Furthermore, as Funk et al. describe in their evaluation of the response to the Syrian conflict, remaining impartial can be impossible. One respondent explained, “You have to understand that even though we declare ourselves as a non-biased health organization with no political standing, the mere fact that we are not ‘pro-government’ makes us [perceived as] ‘the enemy’ and ‘anti-government’” [ 49 ].

The dual imperative

Thirteen articles discuss what humanitarian researchers refer to as the ‘dual imperative,’ which is the inherent tension between ensuring that research is both academically sound and practically relevant [ 28 , 41 , 53 , 55 , 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 71 ]. Despite the inherent challenges in humanitarian research, the general consensus is that it is justifiable insofar as it is needs-driven and not at the expense of humanitarian action [ 60 ]. However, as researchers attempt to construct sophisticated research and attract funding, there is a move toward a greater level of academic sophistication [ 59 ]. On the individual level, a member of a humanitarian response team may feel responsibilities as both service provider and researcher [ 58 , 61 ]. Wood, in her description of experiences researching conflict zones in El Salvador, describes an inevitable self-inquiry of why this research is worth pursing at the expense of a purely humanitarian medical relief mission. She concludes that her role as a researcher was justified in that a sound understanding of conflict is necessary for its abolishment. Wood does, however, concede that this conclusion may be predicated on the nature of the “relatively benign and coherent conditions” of her work. Specifically, she “did not have to make a decision whether or not to intervene to attempt to prevent or mitigate an attack on civilians.” She “did not have to decide how to leave an area under attack at short notice, retreating with one force or seeking shelter from another.” She was “never faced with direct threats [insisting] that [she] turn over material [she] had gathered” and did not have “to judge how far to press respondents about violence they had suffered or observed because of the focus of [her] research.” The implication was that had she been faced with one of these more charged situations, her resolve in the justification of research would be challenged. In fact, she ends her discussion by stating that “conditions in many civil wars simply preclude ethical field research” [ 62 ].

Another related point of contention identified in our search is a disagreement that arose between a researcher and aid agency. Due to an overtaxed and under resourced system, the Democratic Republic of Congo had engaged in rationing of AIDS medications. Rennie, a global health researcher, had intended to study the community attitudes toward this practice [ 55 ]. Feeling rationing medications to be unethical, the aid agency Médecins Sans Frontières (MSF), specifically MSF-Belgium, wrote a letter informing Rennie that they would not support his investigation [ 55 , 63 ]. They expressed concern that the research might be a form of acquiescence to the practice of drug rationing, which they see as antithetical to the humanitarian mission [ 63 ]. This tension between assessing an existing program and unintentionally bringing legitimacy to it is one of many practical conflicts in humanitarian research that requires further consideration.

Clinical trials in the humanitarian setting

Given that clinical trials are considered imperative for investigating medical interventions, many researchers advocate for these types of studies in the humanitarian setting. Thirteen articles explore the ethics of conducting clinical trials in the humanitarian setting [ 27 , 29 , 30 , 36 , 38 , 46 , 51 , 52 , 53 , 54 , 55 , 56 , 63 ]. Lanini et al. make the point that the principle of clinical equipoise should apply in the humanitarian setting as in any other, making randomized controlled trials (RCTs) the most ethical way to conduct research in this situation, using the recent Ebola outbreak and subsequent drug trials to illustrate their point [ 51 ]. With respect to Ebola, Perez et al. make the claim that, given the lethality of the disease, not including pregnant women and children (two groups often excluded from trials on grounds of inherent vulnerability) in Ebola trials is unethical [ 46 ]. This, however, presupposes a benefit to the experimental arm of a hypothetical trial, which would violate the principle of clinical equipoise and thus Lanini et al.’s justification of clinical trials outlined above [ 51 ]. Salerno et al. argue that the unique circumstances of conducting research in humanitarian settings necessitates that the researcher be less stringent in terms of study design. As the authors explain, “the recipients of experimental interventions, locations of studies, and study design should be based on the aim to learn as much as we can as fast as we can without compromising patient care or health worker safety, with active participation of local scientists, and proper consultation with communities” [ 52 ].

Again, with a focus on the recent Ebola outbreak, Calain makes an argument that insistence on RCTs, in which, by definition, one group of participants will be denied the experimental treatment, equates to a preference toward a collective interest (i.e. societal) over the individual (i.e. the patient) which could violate the basic principle of beneficence [ 53 ]. For Calain, in the face of a catastrophic illness like Ebola, randomization of interventions is seen as a “tragic choice” for humanitarian workers [ 53 ]. Furthermore, as Schopper et al. explained, there is justifiable concern that clinical trials during such an epidemic, which require significant amounts of resources and planning, would detract from the crucial work of directly caring for patients in a resource limited setting [ 29 ].

Informed consent

Like formal ethical review, informed consent is another core component of modern research ethics and was separately discussed in ten articles [ 21 , 22 , 23 , 27 , 37 , 38 , 44 , 46 , 65 , 66 ]. Our results highlight several unique considerations when contemplating informed consent in humanitarian settings. For example, Western norms of written consent might be impossible if research is carried out in a population with low literacy rates or when written consent can violate the need for complete anonymity or expeditious research [ 21 , 22 , 44 ]. Controversy surrounding traditional ideas of informed consent were highlighted by Chiumento et al. in their literature review [ 22 ]. The authors explain that despite the general consensus that informed consent was central to ethical research, there were some authors who emphasized a more informal process that considered “consent as a partnership between researchers and participants” [ 22 ]. Some authors surveyed in the study supported flexibility in informed consent by utilizing a “consent framework” that presumably ensures norms such as autonomy and capacity, but allows some latitude for the researcher to adapt to the circumstances. Germane to this point is what Black et al. describe as “dynamic consent”—where a participant’s willingness to be involved in a project is constantly reassessed [ 44 ].

Chiumento et al. explain that because of cultural norms, the typical processes of consent may be undesirable or even impossible [ 21 ]. In their case study of research conducted in a post-conflict setting in South Asia, they explain that the procurement of informed consent first required permission from gatekeepers (i.e. household males and village elders) [ 21 ]. They outline the concept of negotiated consent in which collaboration with researchers helps to distil what exactly culturally specific consent would look like and proceed with an ad-hoc consent process [ 21 ].

Our results suggest that special attention be paid to informed consent during clinical trials conducted in the humanitarian setting [ 29 , 46 , 51 ]. Particularly illustrative is the idea of informed consent for experimental therapies during the Ebola outbreak in West Africa in 2014–2015 [ 46 ]. Authors raise the question as to whether or not informed consent, free of coercion, can really be possible when potential subjects are faced with such a deadly disease [ 23 ].

The use of participatory visual methods (PVM) poses specific challenges with regard to informed consent. The methods ask researchers to encourage subjects to engage in creative forms of communication and expression, such as drama, photography, film, drawing, design, creative writing and music. The products can then be used to engage the community and answer research questions.

However, as participants are synthesizing novel content during the study, and are often encouraged to draw on traumatic experiences as inspiration for this content, fully informed consent is impossible. This is because neither participants nor investigators can completely anticipate which direction their facilitated creative endeavors might turn [ 44 , 65 ]. This type of research may require more creative or dynamic forms of consent such as frequent check-ins with participants, or “dynamic consent”, as described above.

Cultural considerations

The importance of strong appreciation, humility, and understanding of local culture was discussed to a robust degree in six articles [ 21 , 47 , 50 , 57 , 64 , 67 ]. As Black et al. explain, research can only be legitimate if it accepts the people as central actors [ 57 ]. They describe how community and cultural dynamics may be vital to ensuring that the products of research not be utilized in perverse ways [ 57 ]. The authors explain that analyzed and interpreted data on a particular population could be of strategic value to belligerents in a conflict setting [ 57 ]. This notion presents an obvious ethical challenge as it has the potential to make researchers active participants in conflict or surveillance. One may conclude that the solution is for researchers to refuse to share data with any local authorities. This, however, conflicts with what Ditton et al. refer to as a vital aspect of ethical field research, namely “the importance that the researcher has an appropriate relationship with the legitimate gatekeepers [and policy makers] of a field site” [ 47 ]. As the authors note, local authorities may have perfectly legitimate reasons for demanding cooperation and transparency from researchers. For example, in Thailand, government control of researchers might be justifiable since they espouse it as necessary to ensure that the local population is the ultimate beneficiaries of the research produced within their communities. The government, being responsible for the public’s well-being, argues that having some control over research activities is necessary for them to meet this responsibility [ 47 ].

Despite general agreement about the importance of respect for local customs, there is more ambivalence toward which, if any, customs might justifiably be ignored. Bennouna et al. in their survey of researchers explain that 15% of respondents did not believe that local attitudes should be taken into account when deciding on including children in a study, because “what if they tell us not to listen to children?” implying that local norms should not preclude children from having a right to be heard [ 67 ]. In contrast, Chiumento et al. suggest “that ethical conduct of research does not equate to importing cultural norms.” The authors continue to describe a common “ethically charged dilemma” in which consent or access to participants first requires permission from a “gatekeeper.” Cultural norms may dictate that (often male) household or community leaders are to make decisions in terms of participation and access to research, depriving some members of the community of basic “ethic and human rights norms” such as autonomy and the right to participate or refuse [ 21 ]. These points highlight an unanswered question regarding the universality of ethical principles.

Not only might respect for cultural norms be inherently ethically desirable, but it may also be important for ensuring community participation. As Mfutso-Bengo et al. explain, respect for cultural norms may be necessary “to ensure active community involvement as the community does not perceive overt threats to their way of life” [ 50 ]. Balancing fundamental ethical principles of inclusion and autonomy with cultural norms, the articles agree, requires deep cultural understanding.

Risks to researchers

Five of our included articles discuss the potential risk to researchers working in a humanitarian setting [ 21 , 23 , 49 , 68 , 69 ]. With the inherent instability of many of these contexts, Chiumento et al. summarize the wide range of potential risks to the wellbeing of researchers, stating that “threats to physical safety; risk of psychological distress; potential for accusations of improper behavior; and increased exposure to everyday risks such as infectious illnesses or accidents” must be recognized [ 21 ]. The very nature of conducting research in disaster settings exposes researchers to the potential of witnessing “human carnage and physical destructiveness” [ 23 ]. While researchers have personal decision-making responsibilities, host organizations must also acknowledge their obligations to provide security and mitigate risks while ensuring the researchers are fully informed of potential dangers [ 23 , 69 ].

Child participation

Child participation in research was discussed in four articles [ 43 , 65 , 67 , 70 ]. There was a general consensus that despite being particularly vulnerable, researchers had an ethical responsibility to include children in their studies. This action is necessary, the authors conclude, in order to ensure that children’s voices are heard and that they are not excluded from potential benefits of the research [ 67 ].

D’Amico et al. explain “researchers need to develop specific approaches that ensure children understand the benefit of participating voluntarily in research and that consent is informed and an ongoing process” [ 65 ]. The challenge, however, as the authors explain, is that through research, particularly qualitative forms such as PVM, “dangerous emotional terrain” might be breeched [ 65 ]. The implication is that it is difficult to know whether anyone can fully consent to these unforeseen emotional responses, especially children.

Data ownership

Two articles describe the unique ethical concerns surrounding data ownership when conducting research in the humanitarian setting [ 45 , 57 ]. Often, none of the researchers in question are from the communities being studied, so the potential ethical pitfalls of an abusive extractive nature of data collecting might be created [ 45 ]. The concern arises when researchers from high-income countries collect data on lower income communities and the ultimate benefits are seen in the former [ 57 ].

Mental health

Mental health research, which was discussed in two articles, has some unique features, which create special ethical issues [ 21 , 22 ]. For example, Chiumento et al. describe how community mistrust, stigma and paranoia can be particularly significant with regard to mental health, complicating mental health research [ 21 ]. There is also a particular importance for confidentiality and anonymity during mental health research given the potential for discrimination and stigmatizing behavior [ 22 ].

With the drive toward professionalization of humanitarian practice comes a need to develop a strong evidence base. While the latter half of the twentieth century has seen promising trends in favor of ethical standards for research, the unique conditions of humanitarian work and the particular vulnerabilities of the communities being studied makes exploration of humanitarian research ethics imperative. The time-sensitive nature of the work in combination with complex cultural and security dynamics makes conducting research in the humanitarian setting inherently difficult from an ethical perspective.

Efforts to better understand the nexus between research and humanitarian emergencies are expanding. Other research, including an ongoing review of ethics of humanitarian research and more focused analyses of ethics among specific crises will service to expand this knowledge base [ 72 ]. We hope that this paper, representing a broad review and meta-ethnographic analysis of ethical issues in research over more than two decades, strengthens ethical processes and decision making in the humanitarian sector.

Among the 52 articles included in the analysis, 10 major themes regarding the ethics of humanitarian research were extracted for future analysis. In our qualitative analysis of the articles, we found a general acceptance by authors that the increased vulnerabilities of crisis-affected populations lead to several unique issues. Though identified and described in our search, many of these issues have yet to be adequately resolved in a way that might be useful to further researchers. For example, with regard to respect for local cultural norms, our results highlight a unique conflict between a cultural or political demand to share research with a local authoritative body and moral or ethical apprehensions to do so [ 47 , 57 ]. Authors identified both acceptable and unacceptable reasons for an authoritative body to demand access to research [ 47 , 57 ]. The researcher must then decide whether they cooperate with authorities by sharing products of their research, and risk being complicit in less socially desirable actions, or refuse and risk access to their study population, potentially depriving them of the fruits of their work. And to the related point embodied in the disagreement between MSF-Belgium and Rennie, controversy persists as to whether cooperating with an authoritative body to study a practice in which they are engaged suggests support of that practice [ 55 , 63 ]. Further exploration of these questions is essential as the role of research on humanitarian response expands.

Our results suggest that themes of cultural considerations, community engagement and mental health research incorporate ethical dilemmas related to cultural relativism. Accepting cultural norms such as gaining a husband’s consent for his wife’s participation in a research study, or excluding children from a research project on the grounds that including them is too high risk, equates to denying some of the fundamental principles of ethical research. Therefore, researching these populations may mean conceding to certain undesirable cultural norms and rejecting others that would require the researcher to compromise ethical standards. But where should the line be drawn? What guiding principles can future researchers employ? Bennouna et al.’s survey, which revealed most researchers claimed they would, if necessary, ignore local customs and include a child’s point of view in a study might help answer the question [ 67 ]. More of this type of research needs to be done in order to identify and resolve potential conflicts of local norms and traditional research ethics.

A surprising result of our study was that some researchers held the view that certain components of traditional, modern research ethics, such as formal consent, may be applied less rigidly in the humanitarian setting [ 21 , 22 , 44 ]. For example, arguments have been made that any consent is impossible in the case of experimental treatment for Ebola victims, and the failure to meet traditional standards should not preclude one from conducting this research [ 52 ]. On the other hand, there may be certain universal ethical principles of conducting research that should never be compromised. Exactly which principles these are, if any, have yet to be elucidated.

There are further unanswered questions with regard to the involvement of local institutions. Though our results point to a general agreement about the magnanimity of significant local involvement in research, including the development of local capacity for such work the inherent challenges have yet to be addressed [ 27 , 33 ]. Humanitarian research is often conducted in places with little or no infrastructure and limited numbers of qualified researchers. Including local aid workers as researchers, solely for the inherent value of doing so, may prove costly and distract from other research mandates and aid delivery, particularly in disaster relief. As Tansey et al. put it, “while the global health research literature strongly endorses community engagement in all research, there have been few suggestions for overcoming challenges to carrying it out in the disaster setting” [ 33 ]. Future work must come to terms with this inevitable conflict of ideals.

Despite the unavoidable ethical challenges, the results of this systematic review suggest that not only is it possible to conduct research in this context, but there is an ethical obligation to do so [ 41 , 48 ]. If the global community is compelled to provide assistance in the form of humanitarian action, than those in the humanitarian field must acknowledge the responsibility to develop rational, evidence-based approaches that are, at their core, ethically responsible [ 41 ]. This impulse is reflected in our results, which demonstrate an increasing number of publications on humanitarian research ethics since the inception of the Sphere project. The growing body of literature bodes well for researchers looking to ground their future work in a strong ethical foundation.

We would like to note, however, that the vast majority of articles included in this study were from high-income and Western countries. This highlights a finding in the research itself—that community participation and involvement of researchers from the countries and regions affected by crisis is limited. Addressing this inequity should be prioritized as the field of humanitarian research ethics progresses.

It should be noted that our study has limitations. We attempted to conduct a comprehensive review of the literature with a systematic review, augmented by known grey literature, but may have missed some potentially relevant literature that did not fit the search terms and was not identified via the grey literature review. This review is based primarily on published research literature and may exclude operational or programmatic reports with valuable insights. Also, though our initial search did include book chapters via the Scopus database, and dozens of chapters have been written on the subject, relatively few were screened into our final list of included literature. The reason for this is not immediately apparent. The authors did note a relative difficulty in the searching for and screening of book chapters when compared with other types of articles. This may have lead to a preferential selection of the latter type of literature, at the expense of the former.

The selection of papers was systematic and reproducible, and the analysis of those papers relied on standard qualitative methods. While the analysis may be considered less reproducible, we utilized a standardized interpretive methodology that would reliably highlight the critical findings and points within the papers as evidenced by the strong consensus between the authors (WB and RH) on almost every inclusion and exclusion decision. Though the limited literature base makes drawing firm conclusions difficult, the consistency of issues raised between and within the articles confirms the importance of the major themes elicited in this analysis.

This study represents one of only very few attempts at a systematic review of research ethics in the humanitarian setting. We identified an increase in articles with robust ethical discussions particularly in the past few years. This promising trend could lead to further clarification and stronger ethical grounding of future research. Our data also highlight a number of unanswered questions related to fundamental conflicts that are unique to conducting research in the humanitarian setting. There is a clear need for further research and debate addressing these, and other important questions, such as: When is it appropriate to share data with local authorities? At what point should a researcher abandon a cultural relativistic point of view for an absolutist one? In a modern day humanitarian setting, what components of traditional ethics review may be anachronistic? How can researchers include local stakeholders as co-investigators when they may lack the training or infrastructure to do so? Mechanisms to translate these discussions into practical guidelines will need to be strengthened if the ideals of the Sphere Project are to be realized.

Availability of data and materials

The datasets generated and/or analyzed during the current study are available as tables in the manuscript.

Abbreviations

Ethical Review Board

Low and Middle Income Countries

Médecins Sans Frontières

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Acknowledgements

We would like to acknowledge Parveen Parmar and Len Rubenstein for support in developing the conceptual framework of this study.

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This paper proposes a systematic literature review on ethics and CoviD-19, aiming to understand the impact and the perception of the pandemic during the first wave (January-June 2020) and the consequences one year later. PubMed was systematically searched up May 2020 to identify studies that took into consideration various ethical issues that have been arising from the Covid-19 outbreak. The eligibility of the papers was determined by two authors, who screened the results mediated by a third author. In order to facilitate the screening, the titles were divided into five sub-thematic macro-areas, namely allocation , policy , specialist , clinical trials , and technology and, when possible, per geographical area. Specifically, a posteriori, we decided to focus on the papers referring to policies and technology , as they highlighted ethical issues that are not overused and worthy of particular attention. Thus, 38 studies out of 233 met our inclusion criteria and were fully analysed. Accordingly, this review touches on themes such as fairness, equity, transparency of information, the duty of care, racial disparities, the marginalisation of the poor, and privacy and ethical concerns. Overall, it was found that despite the increased awareness of interdisciplinarity and the essential reference to ethics, many scientific articles use it with little competence, considering it only a "humanitarian" enrichment. In fact, as we understand, reflecting a year after the outbreak of the pandemic, although Covid-19 is leading scientists to increasingly recognise the importance of ethical issues, there is still a lot of confusion that could be helped by establishing international guidelines to act as a moral compass in times of crisis.

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1 Background

The spread of COVID-19 begot what the World Health Organisation (WHO) defined a “pandemic” [ 1 ], an emergency condition that has often been compared to other dramatic events in history (e.g., the Spanish flu in 1918). However, on closer inspection, COVID-19 can be regarded as an unprecedented event with its own specificity.

During the first months of the pandemic, the numerous emerging issues were deeply interwoven with ethics, as it is unanimously recognized by the works of politics, medicine, and science. The interdisciplinarity of such works and their continuous and necessary reference to ethics reinforce the belief that bioethics, i.e., a "bridge" between different fields of knowledge, looks more and more "towards the future" [ 2 ]. In fact, in the first few months, the contributions of National Ethics Committees, International Organizations, National Bodies and Professional Association to this topic [ 3 ] were numerous, very rapid, and accompanied a very heated debate on ethical implications between the population and experts from various backgrounds. Nonetheless, the continuous reference to the word “ethics” in the first publications after the declaration of COVID-19 pandemic [ 4 , 5 , 6 , 7 ] highlighted the need to clarify the most relevant ethical problems related to the scientific community. Hence, it was decided to focus on and deepen the subjects that aroused the interest of specialists the most. Specifically, existing works of reconstruction and/or comparison among national and international documents on the relationship between ethics and COVID-19 were not taken into consideration, to avoid delving into or discoursing about already exhaustively discussed topics.

Consequently, a systematic literature review was opted for. Such review was conducted by an interdisciplinary working group, comprising biomedical engineers and bioethicists, following a multidisciplinary approach, which overcomes the outdated Cartesian model of the separation of knowledge into “silos” of disciplines [ 8 , 9 ]. To facilitate the screening process, the papers were grouped by macro areas that were pinpointed through a thematic analysis [ 10 ], as explained in the methods. In particular, it was decided to analyse only the themes related to policy, technology, resource allocation, and low- and middle-income countries (LMICs) as they fall within our competences and seem to underline relevant ethical questions, which are not always adequately or exhaustively discussed. However, the theme of resource allocation was excluded a priori, because it is a trite topic, currently “abused”, on which anyone expresses their opinion independently from their level of expertise and knowledge. Moreover, many papers, which had a reference to ethics in the title, but that, in fact, did not deal with analysing the ethical implications of the investigated issues, were excluded. In fact, the ultimate purpose of this work was to understand the role that scientists recognize in ethics and the problems related to it: is it seen as a “humanitarian” addition to technical issues or as a structural element and perspective from which to analyse specialistic issues?

Therefore, this paper reports a collection of the most relevant and less conventional ethics challenges related to COVID-19 published in peer-reviewed journals indexed in PubMed, analysed through our multidisciplinary lens. These ethical issues, which emerged during the first wave of the pandemic, were then rediscussed a year later, in order to assess whether the first bioethical perspectives related to COVID-19 were biased by the close succession of the events being analysed, or they were detached enough, and the raised issues remain current a year on.

2.1 Systematic literature review

Given the large number of papers and documents that have been and are currently being published since the start of the pandemic, we decided to conduct a systematic literature review. PubMed was selected as the only database to identify all the contributions published from 01/01/2020 up to 19/05/2020 responding to the topics of COVID-19 and ethics. The search string was constructed with the following terms combined with the Boolean operators AND/OR: “ethics”, “ethical”, “bioethics”, “COVID”, “sars cov 2”, “coronavirus” ((ethics OR ethical OR bioethics) AND (COVID OR sars cov 2 OR coronavirus)). We judged eligible only the papers with full text available and in English. In order to facilitate the clustering and further screening of the retrieved articles, they were divided into the most recurring themes and per geographical area. To this regard, the countries were grouped according to the following macro groups, based on the retrieved papers: North America, Europe, LMICs (e.g., Tunisia), Asia, and Israel. Particular attention was dedicated to the contributions regarding LMICs. Finally, only the paper pertaining to our field of expertise, i.e., technologies and policy, made it through the final selection. During this process, two authors independently screened all the titles and abstracts for eligibility. Full texts were considered if the selection was unclear. A third author reviewed and checked the results of the screening search. Any discrepancy was resolved by discussion among all the authors. All the relevant papers were analysed, summarised and coded to facilitate the reading.

3.1 Systematic literature review

Figure  1 shows the electronic database search and the selection process. Only 38 papers out of the initial 233 resulted eligible to be included in our study, which focuses on technology and policy. Figure  2 shows the division in macro groups and Fig.  3 the division per geographical area, when applicable. Online Resource 1 presents all the selected papers organised by recurring themes.

figure 1

The flowchart of the systematic literature review

figure 2

The distribution of the articles in 5 macro-areas

figure 3

The distribution of the articles per geographical area

The 5 pinpointed macro groups of specific works on ethics and COVID-19 were: Policy, Resource allocation, Specialists, Clinical trials, and Technology. The theme of policies, i.e., of the public responses to the crisis, is the one that piques the scientists’ interests the most, according to our review. In this regard, as already mentioned, our review is not exhaustive and does not aim at including all the political guidelines of the various countries, because its hermeneutical horizon [ 11 ] is the point of view of science. Notwithstanding, it was possible to identify a series of specific works on different countries, which allowed comparisons between different areas of the world: LMICs, Asia, Europe, and North America.

The theme of resource allocation is also extremely well-liked. In fact, the first wave of the pandemic caused a scarcity of resources, globally, without any distinction: from personal protective equipment (PPE) to medical devices (MDs) (e.g., ventilators, respirators), beds, drugs for COVID-19 patients or patients suffering from other pathologies, health personnel, and COVID-19 tests. Most specifically, this situation of emergency abruptly showed the lack of competent ad-hoc bodies. This begot extremely heterogeneous approaches to ethics by different individuals, regardless their competence in ethics.

Among the retrieved publications, there were also numerous articles on ethics and COVID-19 written by specialists from various medical fields (e.g., geriatrics, psychiatry, surgery, oncology, and dentistry). Other publication focuses on all healthcare workers (HCWs), such as nurses and midwives, who experienced profound difficulties in this particular historical moment, the common denominator being allocation problems.

A significant number of works was related to clinical trials, in particular concerning vaccines and drugs, dealing with detecting the widespread condition of researchers who had to readdress the scopes of their research, always respecting high ethical standards and safeguarding the testers.

Finally, the last area is that of technology for COVID-19 (e.g., eHealth) and all the related ethical issues.

4 Discussions

4.1 the duality of trust: on the trustworthiness of governments and public trust.

Most of the articles included the topic of infodemic , i.e., “an over-abundance of information – some accurate and some not – that makes it hard for people to find trustworthy sources and reliable guidance when they need it” [ 12 ]. Misinformation circulating through global digital social networks in the first months of this pandemic was focused on trust in governments and policy makers [ 13 , 14 , 15 ], questioning the principles of legitimacy and responsibility related to information verification and sharing.

But who was the source of such disinformation?

Larson [ 15 ] claimed that it is was the governments who repressed information hoping to calm anxious publics, as it happened in Iran [ 15 , 16 ] or in China, where healthcare workers, who told the truth about the spread of the virus and the scarcity of resources, were looked at as “whistle-blowers” and forced to withdraw their declarations [ 17 ]. Moreover, some governments deliberately released supposedly reassuring misinformation, risking undermining their own credibility and their abilities to help people counter real health threats. According to Limaye et al. [ 13 ], there is a need for joint action between government agencies and social media companies for fact-check and even removal of false or outdated information. On the other hand, according to Bastani and Bahrami [ 16 ], there is the need of an active and effective presence of health professionals and authorities on social media, due to the poor legal supervision of online content.

Low-quality research contributed to this misinformation, too. In fact, the high demand for information caused an acceleration in reporting scientific results, with many journals publishing without any peer-review and offering open-access to everyone. A proxy for the high number of low-quality research on COVID-19 is also the unprecedented high number of retracted papers. To this regard, we searched for papers on COVID-19 or SARS-Cov-2 and the previous epidemics/pandemics (i.e., avian flu, swine flu, MERS) both on the Retraction Watch Database and OvidSP. As a result, as of May 2021, 124 out of 264,530 papers on COVID-19 (4.68 retractions per 10,000) were retracted, compared to 1.16 per 10,000 papers concerning the previous pandemics/epidemics.

Apart from being unethical, reporting poor quality outcomes is a kind of research misconduct [ 18 ] and inaccuracy. This could also have other consequences, for example exacerbating stigma and discrimination against particular populations [ 19 ]. In fact, Chowkwanyun and Reed [ 20 ], analysing the information circulated in the media in Wisconsin and Michigan on the high percentage of black people affected by Coronavirus, argued that there is always the need for contextualisation unless we want to foster harmful myths and misunderstanding, which undermine the goal of eliminating health inequities. The fear of stigmatization towards specific groups of people is likely to worsen if they are individuals. In this case, the ethical dilemma is the balance between personal and collective interests.

In hindsight, the public health measures that have been implemented can be of different types. Sulmasy and Veatch [ 21 ] identifies four of them:

Contact tracing through the self-reporting of recent close interactions by people known to be infected with COVID-19. In this regard, Luo et al. [ 22 ] reported the example of an online questionnaire that circulated in China as an internet approach in COVID-19 participatory surveillance.

More "draconian" health measures: new surveillance technologies that employ facial recognition, security cameras, and phone GPS monitoring could attempt to identify everyone who spent at least fifteen minutes within six feet of every infected individual. Each contact could be forcibly quarantined. In this regard, [ 23 ] underlined that apps could also be used as a preventive approach. However, as Vokinger et al. [ 24 ] affirmed, trustworthiness and integrity of contact tracing apps should be assessed with a framework. A review of the tracing apps for the management of COVID-19 can be found in [ 25 ], where the authors underlined that among the advantages there were the increase of personal freedom, of personal feelings of safety, and the improvement of the management of the quarantine. However, governments should implement policies to outline requirements for these apps and should safeguard privacy, access, transparency, the protection and use of these data [ 24 , 25 ]. In particular, Santow [ 26 ] underlined the need for a legal framework to regulate artificial intelligence and data sharing, as they can be the cause of discrimination and violation of human rights.

Voluntary contact tracing: it relies on the self-led contact tracing. However, it is not perfect because patients might not remember all their recent contacts.

The public naming of infected individuals: in this regard, Sulmasy and Veatch [ 21 ] described the case of the Prestigious University, in which the communication via email about the testing positive of a staff member raised a heated debate regarding the request to reveal the identity of the subject in order to maximize public health benefit and slow the spread of the virus. One of the authors argued that this could breach confidentiality and be harmful to the patient's privacy, who is free to decide whether to make a voluntary disclosure. However, another author believed that the confidentiality breach is morally mandatory to decrease the risk of contagion for other members of the University, because the duty towards the community has priority over the right of confidentiality. This is also discussed by Persad and Emanuel [ 27 ], who reported the proposal of some states (i.e., Chile, Germany and the UK) to implement “certifications of immunity” or “immunity passports” for those who had COVID-19 or who will have received the vaccine in the future. In line with the principle of the “least restrictive alternative” to achieve public health objectives, the author believe that this tracking measure is not unethical and cannot be compared to the yellow star that the Nazis forced the Jews to wear, because it is not a form of discrimination. However, it needs careful implementation and scientific support to be ethical in practice.

Another category of individuals who place the interests of society before their own is that of frontline healthcare workers (HCWs) who are forced to work in precarious conditions at their personal risk [ 28 ]. However, for them there is also another ethical dilemma: that of protecting, together with one's own health, also one's family and loved ones, which often clashes with the duty to treat patients, sometimes in the absence of PPE [ 29 , 30 ]. According to McConnell [ 31 ], there are several factors that adjust the burden of protecting one's family. The authors argued that the moral demands of "Samaritanism" (i.e., one should go out of one's way to help someone else, if it entails a little cost to oneself) do not imply that HCWs take on the risks and burdens associated with treating COVID-19 to save several lives. Likewise, Thomas et al. [ 30 ] denounced the low quantity and quality of PPE and the inadequacy of the related guidance issued by Public Health England. Moreover, he appealed to the precautionary principle and praised the ethical framework of Beauchamp and Childress [ 32 ] that encourages to counterbalance beneficence with non-maleficence. Always according to Thomas JP, political leaders have the moral duty to be open and honest, when informing all frontline HCWs of their own personal risks in caring for COVID-19 patients.

Nonetheless, as in all the aspects of life, economical evaluations cannot be excluded. Hilsenrath [ 33 ] presented a very sensitive issue, that of confronting the medical duty to save lives and the reasoning of economists, who invite people to make decisions on appropriate costs. The author lucidly underlined that although the issue is often bypassed by political leaders, especially Americans, it should be faced by considering an ethical balance, certainly painful, between the ethical and economic damage that countries are experiencing in this historical moment.

Overall, what is required from governments is trust. People place their trust, their lives, health, and economic situation in a sensible and transparent decision-making of governments. Therefore, as Thomas et al. claimed, "it is a reasonable expectation to hold our modern governments to the corresponding standards of our modern health professionals: specifically, transparency in decision-making and the duty of candidate" [ 30 ]. Similarly, Lewnard and Lo [ 34 ] sustained that policymakers maintain the public's trust through the use of evidence-based interventions and fully transparent fact-based communication. To give an example of this, the author also reported on mathematical modelling of the viral transmission under different scenarios to generate evidence of the efficacy of social distancing interventions.

On the other hand, Chaari and Golubnitschaja [ 35 ], supporter of 3P (predict, prevent and personalise) medicine, proposed “real-time” monitoring based on randomized laboratory tests as a source of evidence. However, this clashed with the problem of the lack of tests and the discrepancy between officially recorded and real infection cases. In fact, this could lead to incorrect political decisions heavily influencing the future of a country (e.g., a long-term economic recession due to over-protection of the population, or a post-containment pandemic rebound due to an under-protection of the population). Furthermore, all the measures required from governments (i.e., testing, screening, contact tracing, social distancing, travel restriction) must be as inclusive as possible in particular with vulnerable communities (i.e., homeless, those without insurance or employment, disabled, immigrant, prisoners) [ 14 , 34 ]. This also means being aware that the use of technology to combat the spread of COVID-19 could exacerbate racial, socioeconomic and geographic disparities for populations that lack access to reliable internet access, devices or technological literacy [ 36 ]. Moreover, during the pandemic, the benefits of technologies that allow relationships beyond social distancing have increasingly been experienced and could be a valid assistance tool for the elderly [ 37 ] and favour the communication of hospitalised patients with their families [ 36 ]. This response should also involve millennials and Generation Z more [ 38 ], as, despite the negative stereotypes that circulate around them, they could offer valuable help to overcome this crisis.

Certainly, the use of technology, if regulated, can be of help to scientists. Indeed, O'Reilly-Shah et al. [ 39 ] highlighted the shortcomings in the US healthcare IT infrastructures, underlining the importance of the interoperability of healthcare data, which should refrain from the proprietary control of vendors and be accessible to healthcare providers, especially in times of crisis.

More generally, it could be said that technology and data sharing are particularly important for disseminating knowledge worldwide. Momtazmanesh et al. [ 40 ] sustained that sharing and solidarity are at the base of an indispensable international collaboration to fight the current and future pandemics. The pandemic has increasingly shown the need for an international ethical–political coordination framework [ 23 ], aiming at reducing disparities. In this regard, under the category of policy , many contributions regarded LMICs. One of the recurrent themes was the stress on already overburdened and underfunded public healthcare systems in Africa, India, and Latin America [ 41 , 42 , 43 , 44 , 45 ]. In a more general comparison between the North and the South of the world, Schuklenk et al. [ 43 ] affirmed that there is a significant difference in the number of available intensive care unit beds per population and that the access to cures is often wealth-based, as many hospitals in the South are private. However, Krishna [ 42 ] stated that COVID-19 is just another drop in an already full vase, as the Indian healthcare system is already plagued by internal issues (e.g., no access to medicines, vested political interests etc.). All in all, the policies against COVID-19 are, once again, putting the poorest countries and people at risk [ 43 , 46 ]. In India, for example, the poor were confined in ghettos without a proper social security net and their conditions were exacerbated by the lockdown [ 46 ]. Other approaches used in LMICs to fight COVID-19 include the use of fear appeals to regulate people’s behaviours [ 47 ]. However, it can be argued whether this technique is ethical and acceptable or not. On the other hand, Kapata et al. [ 48 ] positively affirmed that Africa was readier than for the previous epidemic outbreaks.

Other diseases, such as vector-borne and non-communicable diseases, and community-acquired infections are often included among other stressors [ 41 , 42 ]. Krishna [ 42 ] also affirmed that notwithstanding this, COVID-19 obtained much more funding compared to other existing deadly diseases (e.g., diarrhoea).

Another element contributing to exacerbating the situation is the lack of ethics committees in the hospitals to regulate and ease the work of healthcare operators. In fact, the latter do not only have to face very difficult choices in an environment dominated by material scarcity and public distrust [ 41 ], but they also have to work in precarious conditions and their striking for this matter could be seen as patient abandonment [ 43 ]. With regard to this, Gopichandran and Subramaniam [ 44 ] recalled the reciprocity principle , according to which the state should protect the interests of the healthcare workers, who risk their lives to care for those who are infected.

Some authors [ 16 , 44 , 45 , 49 ] agreed on the need for equity and clarity in the way governments inform citizens, not to undermine their trust. Most importantly, it was unanimously stated that policies for COVID-19 should always be adapted to different contexts, above all the ones related to minorities, in the respect of traditional beliefs [ 50 ], and to avoid exacerbations of pre-existing gaps between the rich and the poor [ 46 ]. To this purpose, self-determination is key [ 51 ]: in fact, LMICs should independently shape their response, relying on international partners in a critical manner. For instance, LMICs should avoid the unconditional acceptance of measures adopted in high-income countries that would result inappropriate in resource limited settings [ 52 , 53 , 54 ].

Overall, the number and significance of the ethical problems that emerged, which makes us understand how ethics should be increasingly involved in guiding political and scientific legal reasoning, lead us to disagree with Stoeklé and Hervé [ 55 ], who, by separating political discussions, scientific knowledge and ethics writes: "now really isn't the time for ethical reflections" and "ethics is only really useful if you have the time, and right now, time is exactly what we do not have."

5 Conclusions

This paper presented a systematic overview of scientific papers investigating ethical issues during the first wave of this pandemic. The papers highlighted some recurrent themes, namely the allocation of scarce resources, infodemic, HCWs’ duty to treat versus personal protection, privacy, and the safeguard of minorities.

Reviewing such themes, a year after the outbreak of COVID-19, highlights several facts.

The infodemic has been as devastating as the virus. While it could have been acceptable in the first months of this pandemic, it is somehow surprising that after three waves of COVID-19, there is still confusion around trustworthy sources and reliable guidance. A year after, there is still misinformation spread through digital social networks, although the object of the discussion has changed, focusing now on vaccination safety and scarcity, rather than MDs and PPE. The same doubts raised by Larson [ 15 ], Limaye et al. [ 13 ] and Bastani and Bahrami [ 16 ] in March 2020 on the intentional repression of information perpetuated by governments for calming anxious publics during the first wave, have been recently repeated in regard to the surge of COVID-19 that affected India since March 2021.

After more than 12 months from the first wave, the ethical concerns on the stigma and discrimination against particular populations is still relevant in many ways. For instance, several authors hypothesize a causal link between COVID-19 and the surge of violent acts towards Asians living in the USA [ 56 , 57 ]. On the global scale, this discrimination will certainly not be mitigated by the delay in vaccinating populations in LMICs.

Moreover, the ethical concerns arising from balancing the need for track and trace with the risks for privacy, seem to be still unresolved. After a year and half into the pandemic, the same scepticism has crossed many COVID-19 waves resulting in several Apps being abandoned for more traditional phone-based methods. Similarly, the envisioned “real-time” monitoring based on randomized laboratory tests is still far from being possible and could still lead to incorrect political decisions heavily influencing the future of a country. One year on, in fact, although this risk has lowered, globally, it still is critical for some LMICs, where several challenges hinder COVID-19 testing [ 58 ].

Concerns on immunity certifications and pass, are yet far from being resolved, although the attention has now shifted towards the vaccines.

Concerning HCWs, the memory of recognising their efforts by cheering and clapping from the windows is far.

The scarcity of PPE and MDs seems to be overcome now, in many high-income countries, also thanks to the massive effort of United Nation agencies lead by the World Health Organization. Yet, the problem is far from being solved without structural interventions, as demonstrated by the surge in cases in India.

As regards the ethical issues on the appropriateness of economical evaluations and the transparency of political decisions, raised by authors during the first months of this pandemic, there is a need for more time before one can make unbiased reflections.

Inclusiveness seems to be one of the forgotten principles during this pandemic. While this issue was raised from the very start, the question remains open for several groups of populations, which have been not prioritised during the first wave, such as children, poor, unstable workers, chronic patients, and single parents.

Overall, this review identified a number of papers referring to ethics in their title, which in the end presented little or no contribution to the ongoing discussion on ethical issues arising from COVID-19. While the attention of many scholars to this important topic is remarkable, we conclude this review with the doubt that ethics is still considered as a tick-box exercise by many, when not a “humanitarian embellishment” of their technical work.

Conversely, ethics should be deeply interwoven with science, theoretically and methodologically. Besides, helping the evaluation of the arising issues, ethics could help scientists ponder on the risk–benefit balance of their publications, and also on the final purpose of their work, i.e., the progress of humanity. The unprecedented number of retracted papers suggests that during this pandemic the race to participate to the infodemic overcame the sense of responsibility, which should have imposed, in many cases, a responsible silence [ 59 ].

Availability of data and materials

The results of the systematic literature search are available by contacting the authors upon reasonable request.

Abbreviations

World health organization

Low- and middle-income countries

Personal protective equipment

Medical devices

Healthcare workers

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Acknowledgements

DP, AM, LP would like to acknowledge the EPSRC and the Institute of Advanced Studies of the University of Warwick.

DP and LP received support from the University of Warwick with two Warwick Impact Found grants supported by the EPSRC Impact Accelerator Award (EP/K503848/1 and EP/R511808/1). AM’s Fellowship is supported by the WIRL COFUND – Marie Sklodowska Curie Actions, Institute of Advanced Study, University of Warwick (UK). All the above-mentioned funds supported the systematic literature search and review.

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Maccaro, A., Piaggio, D., Pagliara, S. et al. The role of ethics in science: a systematic literature review from the first wave of COVID-19. Health Technol. 11 , 1063–1071 (2021). https://doi.org/10.1007/s12553-021-00570-6

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  • Riemer H J A Slart 31 , 32 ,
  • http://orcid.org/0000-0001-7831-8738 Wolfgang A Schmidt 33
  • 1 Department of Rheumatology and Immunology , Medical University of Graz , Graz , Austria
  • 2 Department of Rheumatology, Teaching Hospital of the Paracelsius Medical University , Brunico Hospital (ASAA-SABES) , Brunico , Italy
  • 3 Department of Rheumatology , Leiden University Medical Center , Leiden , The Netherlands
  • 4 Department of Rheumatology , Zuyderland Medical Centre Heerlen , Heerlen , The Netherlands
  • 5 Department of Rheumatology , Centro Hospitalar Universitario Lisboa Norte EPE , Lisboa , Portugal
  • 6 Rheumatology Research Unit , Instituto de Medicina Molecular , Lisboa , Portugal
  • 7 Leeds Institute for Rheumatic and Musculoskeletal Medicine , University of Leeds , Leeds , UK
  • 8 NIHR Leeds Biomedical Research Centre , Leeds Teaching Hospitals NHS Trust , Leeds , UK
  • 9 Diagnostic and Interventional Radiology , University Medical Center , Wuerzburg , Germany
  • 10 Clinical Department of General Internal Medicine Department, Research Department of Microbiology and Immunology, Laboratory of Clinical Infectious and Inflammatory Disorders , University Hospitals Leuven , Leuven , Belgium
  • 11 General Internal Medicine Department , Universitair Ziekenhuis Gasthuisberg , Leuven , Belgium
  • 12 Department of Medicine , Karolinska Institutet , Stockholm , Sweden
  • 13 Department of Gastroenterology, Dermatology and Rheumatology , Karolinska University Hospital , Stockholm , Sweden
  • 14 Department of Internal Medicine, Division of Rheumatology , Hacettepe Universitesi Tip Fakultesi , Ankara , Turkey
  • 15 Patient Representative , Leicester , UK
  • 16 Department of Autoimmune Diseases , Hospital Clínic, University of Barcelona. Institut d’Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS) , Barcelona , Spain
  • 17 Department of Rheumatology , Hospital General Universitario Gregorio Marañón , Madrid , Spain
  • 18 Rheumatology , Southend University Hospital NHS Foundation Trust , Basildon , UK
  • 19 Anglia Ruskin University , Chelmsford , UK
  • 20 Department of Rheumatology , Aarhus Universitetshospital , Aarhus , Denmark
  • 21 Department of Medicine , Regional Hospital Horsens , Horsens , Denmark
  • 22 Department of Rheumatology , La Paz University Hospital , Madrid , Spain
  • 23 Department of Internal Medicine, Division of Rheumatology , Marmara University School of Medicine , Istanbul , Turkey
  • 24 Department of Internal Medicine, Clinical Division of Internal Medicine II , Medical University of Innsbruck , Innsbruck , Austria
  • 25 Department of Rheumatology , University Medical Centre , Ljubljana , Slovenia
  • 26 Department of Rheumatology , University Medical Centre Ljubljana , Ljubljana , Slovenia
  • 27 Department of Rheumatology , Cochin Hospital, Assistance Publique Hôpitaux de Paris , Paris , France
  • 28 INSERM (U1153) Center of Research in Epidemiology and Statistics (CRESS) , Université Paris-Cité , Paris , France
  • 29 Clinic of Internal Medicine III, Section Rheumatology and Clinical Immunology , University Hospital Bonn , Bonn , Germany
  • 30 Department of Rheumatology and Immunology , Inselspital, University Hospital Bern, University of Bern , Bern , Switzerland
  • 31 Medical Imaging Centre, Department of Nuclear Medicine and Molecular Imaging , University Medical Center , Groningen , The Netherlands
  • 32 Department of Biomedical Photonic Imaging , Universiteit Twente , Enschede , The Netherlands
  • 33 Department of Rheumatology , Immanuel Krankenhaus Berlin, Medical Centre for Rheumatology Berlin-Buch , Berlin , Germany
  • Correspondence to Dr Christian Dejaco, Department of Rheumatology and Immunology, Medical University of Graz, Graz, Steiermark, Austria; christian.dejaco{at}gmx.net

Objectives To update the EULAR recommendations for the use of imaging modalities in primary large vessel vasculitis (LVV).

Methods A systematic literature review update was performed to retrieve new evidence on ultrasound, MRI, CT and [ 18 F]-fluorodeoxyglucose positron emission tomography (FDG-PET) for diagnosis, monitoring and outcome prediction in LVV. The task force consisted of 24 physicians, health professionals and patients from 14 countries. The recommendations were updated based on evidence and expert opinion, iterating until voting indicated consensus. The level of agreement was determined by anonymous votes.

Results Three overarching principles and eight recommendations were agreed. Compared to the 2018 version, ultrasound is now recommended as first-line imaging test in all patients with suspected giant cell arteritis, and axillary arteries should be included in the standard examination. As an alternative to ultrasound, cranial and extracranial arteries can be examined by FDG-PET or MRI. For Takayasu arteritis, MRI is the preferred imaging modality; FDG-PET, CT or ultrasound are alternatives. Although imaging is not routinely recommended for follow-up, ultrasound, FDG-PET or MRI may be used for assessing vessel abnormalities in LVV patients with suspected relapse, particularly when laboratory markers of inflammation are unreliable. MR-angiography, CT-angiography or ultrasound may be used for long-term monitoring of structural damage, particularly at sites of preceding vascular inflammation.

Conclusions The 2023 EULAR recommendations provide up-to-date guidance for the role of imaging in the diagnosis and assessment of patients with LVV.

  • Giant Cell Arteritis
  • Ultrasonography
  • Magnetic Resonance Imaging

https://doi.org/10.1136/ard-2023-224543

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Introduction

Giant cell arteritis (GCA) and Takayasu arteritis (TAK) are the most common primary large vessel vasculitides (LVV). 1 2 Temporal artery biopsy (TAB) and conventional angiography have been considered for decades the ‘golden standards’ for diagnosing GCA and TAK, respectively. In the last years, however, imaging modalities including ultrasound, MRI and [ 18 F]-fluorodeoxyglucose positron emission tomography (FDG-PET) have become an integral part of the diagnosis, supported by the 2018 EULAR recommendations for the use of imaging in LVV. 3

Since 2018, new studies on imaging in LVV have been published, some of them prompting a reconsideration of the original statements. FDG-PET, for example, was previously considered inadequate for the assessment of temporal arteries because of the proximity to the brain. 3 However, recent studies report that FDG-PET can detect temporal arteritis with high sensitivity and specificity. 4–6

Glucocorticoids (GC) are the mainstay in the treatment of GCA and TAK, but recently, the interleukin-6 receptor (IL-6R) inhibitor tocilizumab has become part of the standard treatment for GCA. 7 Studies indicate that this drug is also effective in TAK, although approval for this indication by the Food and Drug Administration (FDA) and the European Medicines Agency is still lacking. 8 The IL-6 R inhibitor sarilumab has been approved by FDA for Polymyalgia rheumatica (PMR), a disease that is considered to be part of the GCA-PMR spectrum. 9 10 These agents interfere with the ability to produce an acute phase response as evaluated by erythrocyte sedimentation rate (ESR) or C reactive protein (CRP), reducing the impact of these biomarkers on clinical assessment of disease activity in LVV. Imaging modalities therefore may contribute to a more comprehensive assessment of patients with GCA and TAK. 11

The American College of Rheumatology (ACR) has recently published management guidelines for GCA expressing their preference for TAB over ultrasound and MRI of cranial arteries in the diagnosis of GCA, which contrasts with the 2018 EULAR recommendations 12 and other recommendations. 13–16 The ACR clarifies that these recommendations relate in part to the lack of technical expertise of clinicians in the USA with these modalities. 12

These and other developments have prompted us to re-evaluate and update the original EULAR recommendations, particularly addressing uncertainties about the choice of the imaging technique for diagnosis and assessment of patients with GCA and TAK. The objectives, target population and target users of this update remain unchanged compared with the previous version. 3 These recommendations are intended to advise physicians on the use of imaging modalities in patients with suspected or established primary LVV, specifically GCA and TAK.

After approval by the EULAR Council, the convenors (CDejaco and WAS) and the methodologist (SR) led a task force guided by the 2014 updated EULAR Standardised Operating Procedures (SOP), complying also with the principles stipulated in the 2022 EULAR SOPs. 17 18 The 24 task force members from 14 countries consisted of rheumatologists, a radiologist, nuclear medicine specialists, an internist, epidemiologists, a patient representative (the second patient could not attend the meeting for health reasons), a health professional in rheumatology and 2 EMerging EUlar NETwork representatives. Five members were recruited through an open call to EULAR countries via a competitive application process. All members disclosed their potential conflicts of interest before starting the process. A single face-to-face task force meeting took place. In preparation of that meeting, several online conferences of the steering committee (CDejaco, SR, WAS, MB, PB, CP and SLM) were conducted. For the systematic literature review (SLR), we used the same Population, Intervention, Control, Oucome (PICO) questions and inclusion/exclusion criteria as in the original project, 19 covering the role of ultrasound, MRI, CT, FDG-PET in the (1) diagnosis and (2) monitoring of inflammation and damage, (3) prediction of outcome and (4) required technical standards for imaging. The steering committee made a single modification of the original PICO questions including optical coherence tomography and fluorescein angiography (FA) as additional imaging techniques in the search.

The SLR was conducted by two fellows (MB and PB) under the guidance of the methodologist (SR). Papers published between March 2017 and 16 November 2022 were considered. Some articles were identified by both the original 19 and the present SLR and were therefore excluded manually from the current update. Only prospective and cross-sectional studies conducted in >20 adult patients with suspected and/or established GCA or TAK were included. We chose a cut-off of 20, because studies with a lower number of participants were concerned to be of lower quality and would therefore be less relevant for the task force. The evidence summarised in the SLR was presented to the task force in the form of tables summarising the findings, including an assessment of the risk of bias (RoB). 20 21 The evidence collected in the previous SLR was also considered and summarised to the task force. 19 Given the large number of studies included in the complete SLR (original and current update), a conscious choice was made to favour evidence stemming from studies with low RoB. The present SLR is published separately 11 ; however, the SLR and the present recommendations manuscript form an integral and inseparable part and should be read as such.

Based on new evidence and expert opinion, the steering committee prepared proposals for updated recommendations which were subsequently discussed and refined by the entire group during the task force meeting. Following the EULAR SOP, a modification of the original statements had to be approved by >75% of the task force members, followed by a second voting on the final wording of the rephrased recommendation. 18 Consensus was accepted if >75% of the members voted in favour of the statement in the first round, >66% in the second round and in the third round >50% was accepted. The 2011 Oxford Centre for Evidence Based Medicine levels of evidence (LoE) derived from the SLR were added to each recommendation. 22

Finally, each task force member anonymously indicated the level of agreement via Survey Monkey (LoA, 0–10 numeric rating scale with 0=do not agree and 10=fully agree). The mean and SD of the LoA, as well as the percentage of task force members with an agreement ≥8 are presented.

The task force specified that CT and MRI also refer to the specific angiography techniques such as CT-angiography (CTA) and MR-angiography (MRA), respectively, and PET is commonly used in conjunction with CT or CTA, and occasionally in combination with MRI/MRA.

The original research agenda was checked as to whether any of the gaps in evidence have meanwhile been filled with evidence. 3 New issues arising during the task force meeting were added to the research agenda presented in this paper. The final manuscript was reviewed and approved by all task force members and by the EULAR Council.

General aspects

In 2018, 12 recommendations were formulated, while the update includes three overarching principles and eight specific recommendations. Compared with the original statements, two (new numbers 5+6) remained unchanged, two underwent minor (4+8) and four major modifications (1–3, 7). The former recommendation five on CT and FDG-PET for the assessment of cranial arteries was deleted (see online supplemental table 1 for a comparison between the original and the updated statements).

Supplemental material

The updated recommendations are depicted in table 1 (including the LoE and LoA) and are explained in detail below.

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2023 EULAR recommendations for the use of imaging in large vessel vasculitis in clinical practice

Overarching principles

Overarching principles are statements of generic nature and are not necessarily based on direct evidence. They reflect principles of good clinical practice and build the framework for the subsequent specific recommendations.

Overarching principle A

In patients with suspected GCA, an early imaging test is recommended to support the clinical diagnosis of GCA, assuming high expertise and prompt availability of the imaging technique. Imaging should not delay initiation of treatment.

This statement remained largely unchanged compared with the original recommendation. 3 The task force emphasised that an imaging test should be considered in every patient with suspected GCA. TAB is also an adequate option, particularly when imaging is not readily available or expertise with imaging in GCA is insufficient. It is commonly believed that operator dependency is the major limitation of imaging, particularly of ultrasound. However, all diagnostic procedures including TAB are subject to operator dependency; and reproducibility can be improved by specific training. 23 24 In addition, a multicentre study including sonographers with a mixed level of experience revealed that inter-reader reliability of ultrasound images was comparable to that of digital histological images. 25

The main point of discussion within the task force was whether treatment could be delayed while waiting for imaging to be done. It was felt that patients with strongly suspected GCA, particularly if there are ischaemic manifestations of concern (eg, transient visual loss or jaw claudication), must be treated immediately due to the risk of imminent and permanent visual loss. Blindness and other ischaemic complications of GCA tend to occur almost exclusively before initiation of GC therapy. 26 For patients presenting with polymyalgia or systemic symptoms only and in whom GCA is a possible diagnosis, FDG-PET and MRI to detect large vessel involvement might not be available immediately when requested. One study demonstrated that the sensitivity of a FDG-PET for the diagnosis of GCA conducted 72 hours after the start of 60 mg prednisone remained equal to the pretreatment FDG-PET, but dropped to 36% after 10 days of GCs. 27 Similarly, ultrasound of temporal arteries performed within 1 day of GC therapy revealed a significantly higher intima-media thickness (IMT) than scans performed (in different patients) after 1 week. 28 The task force, therefore, concluded that imaging should best be performed before, or at least, within 72 hours after commencing GC therapy.

The task force urged rapid access to diagnostic imaging tests, commensurate with fast-track clinics for patients with suspected GCA. 29–31 While ultrasound has become a point-of-care tool in many of these fast-track services, long waiting lists are still a major limitation of other imaging modalities. The task force was reluctant to define case scenarios where treatment could be withheld until imaging becomes available. This would not only be counterproductive for the development of rapid access pathways for imaging, but would also expose people with GCA to an unnecessary risk of blindness and other ischaemic manifestations, given that not all patients who develop GCA-related visual loss have characteristic symptoms of cranial GCA. 32

Overarching principle B

Imaging examination should be done by a trained specialist using appropriate equipment, standardised operational procedures and settings.

Specific training in vasculitis imaging, rather than general training in musculoskeletal or vascular imaging, is pivotal to guarantee high-quality results. 24 A dedicated training curriculum for rheumatologists still has to be defined. According to expert opinion such a curriculum should include teaching about the signs and symptoms of LVV (in order to appropriately estimate the pretest probability of the disease), specific training in vascular ultrasound as well as instructions on how to correctly interpret the results of additional tests, including other imaging modalities. Recommended technical and operational parameters are detailed in box 1 .

Suggestions for technical and operational parameters on imaging modalities in large vessel vasculitis

High-quality modern equipment is essential. Linear probes are recommended for supra-aortic arteries; sector or convex probes for ascending aorta; convex, sector or linear probes for the aortic arch; and convex or linear probes for abdominal aorta. Settings may vary slightly according to equipment.

The B-mode frequency should be ≥15 MHz (preferably ≥18 MHz) for temporal arteries and 7–15 MHz for extracranial supra-aortic arteries. Image depth should be 10–20 mm for temporal arteries and 30–40 mm for extracranial supra-aortic arteries.

The focus should be at the level of the artery. The B-mode gain should be adjusted to avoid anechoic appearance of the artery wall.

Colour Doppler is the preferred modality to show the blood flow, but other techniques may be used as well. The gain should be adjusted to avoid underfilling or overfilling of the vessel lumen.

Doppler frequency and pulse repetition frequency (PRF) settings depend on the equipment. Frequencies of 7–12 MHz and 4–8 MHz as well as PRFs of 2–7 KHz and 3–8 KHz are commonly applied for temporal and extracranial supra-aortic arteries, respectively. The colour box should be angled in longitudinal scans to avoid perpendicularity between sound waves and artery.

Multislice CT-scanner should be used.

Collimation 0.6 mm, individual tube voltage and tube current time product determined by automatic dose modulation.

Reconstruction slice thickness should be between 0.5 and 1.0 mm.

Patient and CT technology adapted injection of 50–100 mL of non-ionic iodinated contrast agent (≥350 mg/mL) using a power injector (≥4 mL/s).

Arterial phase: bolus-tracking method (threshold of 100 HU); ECG triggering.

Venous phase: 50 s after finishing the arterial phase acquisition.

Angulated reconstructions for assessment of individual vessel segments.

Cranial MRI technique ** :

3.0T MRI scanner, minimum 16-channel head-coil.

T1w Spin Echo, Gadolinium contrast enhanced, fat-suppressed, high-resolution (in-plane<1 mm 2 , eg, 195×260 µm 2 , slice thickness 3 mm, TR/TE 500/22 ms).

Transversal slices angulated parallel to skull base.

Reformatted 3D contrast-enhanced vessel-wall MRI may be considered.

Body MRI technique:

1.5 T, preferentially 3.0 T MRI scanner, minimum 16-channel head and neck coil and 16-channel body-coil.

MR-angiography (MRA) of aorta and major branches from carotid bifurcation to abdominal and preferentially pelvic arteries in coronal acquisition to include axillary and brachial arteries → detection of vessel lumen (stenosis, occlusion, aneurysm).

T1w, fat-suppressed, contrast enhanced, black blood imaging (eg, navigated 3D Turbo Spin Echo (TSE), spatial resolution 1.2×1.3×2 mm 3 , Repetition Time (TR) / Echo Time (TE) 1000/35 ms) → assessment of mural inflammation.

[ 18 F]-fluorodeoxyglucose positron emission tomography (FDG-PET) ***

Position of patient is supine, position of the arms should be arms down.

Body parts to include: from top of head to at least mid-thigh, preferably to below the knees.

Blood glucose levels: preferred <7 mmol/L (126 mg/dL), <10 mmol/L (180 mg/dL) acceptable.

Interval between FDG infusion and image acquisition should be at least 60 min, preferably 90–120 min.

For evaluation of the cranial arteries, 5 min instead of 2–3 min acquisition time of the head should be used in case of non-digital FDG-PET imaging.

Scoring of [ 18 F]-FDG-uptake: qualitative visual grading; if result is unclear, compare it to the liver background (grading 0–3).

Digital FDG-PET may be used in order to reduce imaging time, radiation dose and to improve the image quality.

FDG-PET is commonly combined with low-dose CT, optionally with CT-angiography (CTA). It can also be combined with MRI or MRA.

* CT and MRI also refer to specific angiography techniques such as CTA and MRA.

** MRI of the head can visualise superficial temporal, posterior auricular, superficial occipital branches and vertebral arteries.

*** FDG-PET of the head can visualise superficial temporal, facial, maxillary, superficial occipital branches and vertebral arteries.

Overarching principle C

In patients in whom there is a high clinical suspicion of GCA and a positive imaging result, the diagnosis of GCA may be made without an additional test (biopsy or further imaging). In patients with a low clinical probability and a negative imaging result, the diagnosis of GCA can be considered unlikely. In all other situations (including the case of an inconclusive imaging result), additional efforts towards a diagnosis are necessary.

This statement is unchanged from the 2018 version. 3 It remains at the discretion of the clinician to determine the pretest probability for GCA, considering all available clinical and laboratory data. The precision of the pretest estimates depends largely on the experience of the clinician, which may (similar to imaging) be improved by specific training. A probability score combining demographic, clinical and laboratory parameters, as well as respective cut-offs defining low, intermediate and high probability for GCA has been proposed and initially validated by several centres 33–35 ; however, this awaits further experience.

The present recommendation emphasises the dependence of the (post-test) diagnosis on the pretest probability and the imaging test result. While in clinical practice, a global report of the imaging exam is usually provided (‘positive’/‘negative’), the confidence in this result may vary. For instance, there is a lower confidence in a positive result for patients in whom a limited vessel wall thickening was documented compared with those with extensive swelling of several vessels. If imaging results are inconclusive, such as when wall thickness is only slightly above the normal cut-off or when arteriosclerosis complicates the assessment, additional testing is necessary to establish or exclude GCA. This is also the case if positive imaging occurs in situations with low pretest probability. Additional testing is also important if GCA mimickers are suspected. 36 In some diseases such as granulomatosis with polyangiitis, lymphoma or amyloidosis, imaging abnormalities similar to those found in GCA have been described; relevant differential diagnoses need therefore to be excluded on clinical grounds, by laboratory exams and/or histology. 37–40

Recommendations

Recommendation 1.

Ultrasound of temporal and axillary arteries should be considered as the first imaging modality to investigate mural inflammatory changes in patients with suspected GCA.

Ultrasound is the first imaging modality for the diagnosis of GCA because of strong evidence for a high diagnostic value, with a pooled sensitivity from studies with low RoB of 88% and a specificity of 96%, using clinical diagnosis as the reference standard. 11 Furthermore, ultrasound is known for low resource consumption, easy and prompt availability in many institutions and the absence of radiation. Reliability of ultrasound findings has been high among trained experts, 24 41 and it was comparable to that of reading biopsies by pathologists. 25 The task force chose the term ‘should be considered’ rather than ‘should be used’ acknowledging that rapid conduction of ultrasound is not feasible in every setting, particularly in non-tertiary hospitals, and that not all specialists diagnosing GCA have sufficient training in this technique. In these situations, other imaging techniques can be used (see outlined below) and TAB is also an adequate alternative. 3

While in 2018, a distinction was made between cranial and extracranial phenotypes, and ultrasound was recommended in first place for the former subset, 3 the task force now recognised that GCA represents a single disease spectrum with overlapping phenotypes. 42 Overall, 35%–80% of patients with dominant ‘cranial’ manifestations have large vessel involvement in imaging studies, and patients with predominantly ‘extracranial’ manifestations at disease onset might develop cranial disease at later stages. 43–45 The assessment of axillary arteries is now recommended for every patient with suspected GCA as it increases the diagnostic yield and can serve as a baseline for the comparison with possible future examinations. Studies (particularly with low RoB) investigating both temporal and axillary arteries revealed consistently higher sensitivities than those focusing on temporal arteries only. 11 In situations where a scan of temporal and axillary arteries is non-diagnostic, and the clinical suspicion for GCA is still high, additional vessels such as facial, occipital, carotid, vertebral, subclavian and femoral arteries as well as the aorta can be investigated. 46–49

Ultrasound composite scores have recently been proposed for diagnostic and monitoring purposes in GCA. 50 51 One of these, the Southend score, positively correlated with the diagnostic specificity for GCA, and patients with higher scores were more likely to have positive TAB and visual complications than patients with lower scores. 50 52 The provisional OMERACT ultrasonography score (OGUS) has been developed for the follow-up of patients in research studies and will be described below. 51 Despite these interesting developments, the task force was reluctant to recommend any ultrasound composite score at this stage outside the research setting, encouraging their further validation.

The broad description ‘mural inflammatory changes’ was used in the update, rather than the term ‘non-compressible halo sign’ stipulated in the original recommendations. 3 The compression sign for axillary and other extracranial arteries has not been tested in clinical studies so far, and patients with a new diagnosis of GCA may reveal signs of chronic vasculitis (eg, when GCA is newly diagnosed in a patient with long-standing PMR). The ultrasound appearance of chronic vasculitis has recently been described as ‘mid-hyperechoic wall thickening with a multilinear pattern’ that can clearly be distinguished from the ‘halo’ sign that is described as homogenous, hypoechoic wall thickening reflecting acute inflammation. 53 54 Cut-off values for the thickness of the intima-media complex have been proposed for follow-up studies (common superficial temporal artery 0.42–0.44 mm, parietal branch 0.29–0.36 mm, frontal branch 0.34 mm, axillary artery 1.0 mm (0.9 mm for chronic vasculitis)). 55–58 They may also aid in distinguishing normal from acute or chronic vasculitis; however, further studies are required to validate these values, particularly in people with advanced age or with severe arteriosclerosis. 59–61

Recommendation 2

High-resolution MRI or FDG-PET can be used as alternatives to ultrasound for the assessment of cranial arteries in patients with suspected GCA.

In the original recommendation, 3 the use of FDG-PET and CT were discouraged for the assessment of cranial arteries as there was insufficient evidence to suggest that these vessels were visible by these techniques. Since then, a number of studies supported the utility of FDG-PET for the diagnosis of temporal arteritis, and clinical experience indicates the potential visualisation of superficial cranial vessels by CT. 4 5 62 This technique, however, was not included in the updated recommendation because of the absence of prospective data.

The task force decided to maintain separate recommendations for cranial and extracranial arteries concerning MRI and FDG-PET. The evidence supports the use of high-resolution MRI for cranial arteries, with a pooled sensitivity of 81% and a specificity of 98% (studies with low RoB) using clinical diagnosis as reference standard, 11 while for FDG-PET, studies were mainly conducted to assess extracranial (and only to a lesser extent cranial) vessels. FDG-PET of cranial and extracranial arteries yielded a pooled sensitivity of 76% and a specificity of 95% (studies with low RoB) applying clinical diagnosis as reference standard. 11 The advantages of MRI and FDG-PET over ultrasound are a higher standardisation of data acquisition and the possibility to investigate multiple vessels at the same time. The main limitations are the restricted availability, high costs and possible adverse effects of contrast agents (MRI) and radiation (FDG-PET). 63

It could be argued that MRI should still be preferred over FDG-PET to image cranial vessels given that at this site, MRI is supported by a higher number of studies. In addition, MRI does not imply radiation and, in some centres, it is available at lower costs. 11 Rapid availability of MRI, as specified in our overarching principles, is still restricted to a small number of centres with a special interest in the field. However, our recommendations might encourage the development of fast-track imaging pathways, including MRI or FDG-PET organised within a few days, ideally within 72 hours of commencing GC therapy. 27

Recommendation 3

FDG-PET, alternatively MRI or CT, can be used for the detection of mural inflammation or luminal changes of extracranial arteries in patients with suspected GCA.

FDG-PET should be considered the first imaging alternative to ultrasound of extracranial vessels because of evidence supporting a high diagnostic value (sensitivity 76%, specificity 95%, clinical diagnosis as reference standard), 11 and the possibility of detecting other serious pathologies such as infections or tumours, particularly in patients with atypical symptoms. 5 Although prospective evidence for the performance of MRI and CT of extracranial vessels is scarce, 19 some members of the task force reported their clinical experience that these techniques may be valid alternatives to ultrasound and FDG-PET when these are not available or inadequate. We intentionally listed CT as last in the list of recommended imaging modalities because there is no evidence that CT performs better than MRI in this indication, but exposes the patient to radiation. 64 On the other hand, there might be practical reasons why CT is preferred in certain situations such as the lower acquisition time and shorter waiting lists.

Direct comparisons between imaging modalities are scarce. One study reported that ultrasound of temporal arteries revealed a higher sensitivity than FDG-PET, while FDG-PET performed better at subclavian and carotid arteries. 65 At axillary arteries, ultrasound detected 76% of cases with vasculitis using FDG-PET as reference. 66 Another study concluded that in patients with LVV, FDG-PET better captures inflammatory activity, while MRI is better suited to assess disease extent. 67 These studies were not included in the SLR because of their case–control design, and their results need to be confirmed by prospective cohort studies before conclusions can be applied to clinical practice.

FDG-PET is usually combined with low-dose CT (or CTA) and optionally with MRI/MRA. 68 The evaluation of luminal changes is therefore possible with FDG-PET, however, MRA or CTA (without FDG-PET) may be sufficient for this indication. 67 A prospective comparison between MRA and CTA for the evaluation of arterial stenoses or aneurysms in GCA has not been conducted so far, and therefore, this aspect has been added to the research agenda.

Recommendation 4

In patients with suspected TAK, MRI to investigate mural inflammation or luminal changes should be used as the first imaging test to make a diagnosis of TAK.

Recommendation 5

FDG-PET, CT or ultrasound may be used as alternative imaging modalities in patients with suspected TAK. Ultrasound is of limited value for assessment of the thoracic aorta.

Recommendation 6

Conventional angiography is not recommended for the diagnosis of GCA or TAK as it has been superseded by the previously mentioned imaging modalities.

Recommendations 4–6, which concern the use of imaging methodologies for the diagnosis of TAK, remain largely unchanged compared with the 2018 version given the absence of new prospective data. 3 MRI is preferred over the other imaging modalities for the diagnosis of TAK because of the absence of radiation (taking into account that serial imaging is required in many patients) and because of the ability to investigate several vessels simultaneously, including the aorta. The descending thoracic aorta, which is frequently affected in patients with TAK, is not accessible to conventional ultrasound. Whether transoesophageal echocardiography, enabling a comprehensive assessment of the thoracic aorta, is helpful in these patients needs to be studied further. 69 The task force emphasised that conventional angiography is indicated for vascular interventions rather than for diagnosis. It should be conducted in a quiescent phase of the disease. 7

Recommendation 7

In case of a suspected relapse, particularly when laboratory markers of disease activity are unreliable, ultrasound, FDG-PET or alternatively MRI may be considered for the assessment of vessel abnormalities. Imaging is not routinely recommended for patients in clinical and biochemical remission.

Since the publication of the 2018 recommendations, several new studies have become available investigating the value of ultrasound and FDG-PET for the assessment of GCA patients during follow-up, and to a lesser extent of patients with TAK. 11 Two major ultrasound composite scores have been developed, the Southend score and the OGUS. Both include eight arterial segments (bilateral common, frontal and parietal branches of temporal arteries and axillary arteries) with semiquantitative (0–3) or quantitative scoring of each segment, respectively. 50 51 For FDG-PET, the PET Vascular Activity Score or Total Vascular Score have mostly been used, which evaluate nine (ascending, descending thoracic and abdominal aorta, aortic arch, brachiocephalic, bilateral carotids and subclavian arteries) or twelve (thoracic aorta, abdominal aorta, subclavian arteries, axillary arteries, carotid arteries, iliac arteries and femoral arteries) vascular territories, respectively, on a semiquantitative scale (0–3). 70 71 Most studies applying these imaging composite scores revealed a good sensitivity to change as well as moderate correlations with markers of disease activity. 11 72 No new studies were available for MRI in this regard.

Despite these important advances, the task force concluded that evidence is not strong enough to recommend imaging-based follow-up assessment of inflammation in all patients with LVV. Particularly, there are no studies demonstrating the added value of regular imaging of patients with GCA or TAK over clinical and laboratory monitoring alone. A specific situation in which imaging can be useful is the case of non-specific symptoms with increased inflammatory markers, or the occurrence of a new ischaemic event/worsening of disease-related ischemia. Another example is the evaluation of a possible relapse in patients treated with drugs blocking the interleukin-6 pathway, given that ESR and CRP are not clinically informative in these patients. The GUSTO trial, not included in the SLR because <20 patients were recruited, demonstrated an improvement of the IMT assessed by ultrasound after GC pulse therapy and subsequent worsening of the IMT once GCs were stopped. 73 74 During treatment with tocilizumab, which was started immediately after GC pulses, IMT gradually decreased.

Another indication for imaging is the identification of the disease subtype in TAK. 75 During the follow-up of patients with GCA or TAK, imaging could be used for the stratification of disease such as the identification of the number of vessels involved (including the aorta) and the graduation of inflammation at the single vessel, particularly in patients with refractory or relapsing disease. 42 An imaging composite score might be valuable for this purpose; however, all available imaging-based scores require further validation. Besides, imaging scores are used in a research setting, but have not yet been demonstrated to add value in daily clinical practice. 50 51 76–78

Imaging is not routinely recommended to evaluate vascular inflammation in patients in clinical remission. Nevertheless, the interpretation of imaging results obtained at the time of a suspected relapse can be facilitated by the comparison with a previous imaging examination, which also includes an assessment conducted at the time of inactive disease. The main reason for the reluctance of the task force to recommend imaging in patients in remission is the unclear clinical significance of persistent imaging abnormalities (reported by several studies), and whether these should lead to a change in treatment. 70 77 78 Some studies indicate that the risk of a clinical relapse is associated with the intensity of FDG-PET uptake in remission, 71 however, others did not confirm these findings. 70 79

Another point of discussion was whether worsening of existing or new damage (eg, a new stenosis) may support the clinical suspicion of relapse. While this might sound intuitive, the question of whether pre-existing damage might progress independently of active inflammation is unclear yet; hence, this aspect has been added to the research agenda.

Recommendation 8

In patients with GCA or TAK, MRA, CTA or ultrasound of extracranial vessels may be used for long-term monitoring of structural damage, particularly at sites of preceding vascular inflammation. The frequency of screening as well as the imaging method applied should be decided on an individual basis.

The key modification of this recommendation is the incorporation of the concept that vascular damage mainly occurs at sites of preceding vascular inflammation. 3 This suggestion is based on the results of one study of 32 GCA and 28 TAK patients, reporting that 80% of territories subsequently developing angiographic changes (stenosis or aneurysms) during follow-up displayed a significant FDG-uptake at baseline. 80 On the other hand, 92% of territories with baseline FDG-PET activity did not yield angiographic changes over time. The present statement should therefore not be understood as a recommendation to perform vascular screening by FDG-PET in every GCA or TAK patient at baseline given the absence of data on the cost-effectiveness of such an approach.

The term ‘vascular damage’ relates to all types of vascular structural deformities identified by imaging including stenosis, occlusion, dilatation and/or aneurysms, as well as arteriosclerosis and fibrosis. Aortic dilatation appears to be more common in patients with GCA than in the general population. 81 82 Mortality in GCA is increased in patients with aortic aneurysm and dissection as compared with those without. 83 Risk factors for aortic dilatation are male sex, hypertension, smoking history and imaging evidence of aortitis according to some studies, 81 84 while others found aneurysms more frequently among females aged under 70 years and positive TAB. 82 Despite the increased risk of aneurysm development, it is still unclear how many GCA patients need to be screened to prevent one additional rupture/dissection or to detect one additional aneurysm requiring surgery. 85–87 Another point of uncertainty is the frequency and minimum duration of screening, given that aortic dilatation may develop several years after GCA onset despite prolonged clinical remission. 82 87 Routine assessment of all patients for damage is therefore not recommended; the task force suggests, as in 2018, to screen patients with signs or symptoms of stenosis/occlusion or aneurysms for damage, as well as those with recurrent or persistent inflammation of large arteries including the aorta.

No recommendation was made on the value of imaging modalities for outcome prediction of patients with established GCA. The SLR retrieved five studies (three on ultrasound, two on FDG-PET) indicating that baseline imaging did neither predict response to treatment, nor occurrence of relapses or ischaemic complications. 11

None of the items listed in the research agenda published in 2018 has been sufficiently filled with evidence. 3 Based on the points of discussion during the current task force meeting, a research agenda has been proposed addressing new aspects that have emerged since the publication of the original imaging recommendations (see box 2 ).

Research agenda

To investigate and compare the value of imaging composite scores (total scores, (semi)quantitative (cut-off) scores, number of involved vessels, cranial vs extracranial vessels) for diagnosis, monitoring and prognosis of giant cell arteritis (GCA) and Takayasu arteritis (TAK).

To investigate the prognostic value of positive imaging findings in patients in clinical and biochemical remission, value of repeated imaging for comparison of imaging findings in remission and relapse.

To better investigate the added value of ‘optical coherence tomography’ and ‘fluorescein angiography’ for diagnosis and prognosis of GCA.

To investigate the value of ultrasound of all supra-aortic vessels as compared with the assessment of the axillary artery only, in addition to the temporal arteries.

To investigate the value of assessing the aorta for vasculitic involvement in patients with imaging evidence of cranial vasculitis versus extracranial vasculitis.

To investigate the timing at which imaging should be conducted to detect vessel wall damage.

To study the role of artificial intelligence in the assessment of large vessel vasculitis by various imaging methods.

To study positron emission tomography (PET) with novel tracers such as ligands targeting immune cells.

To study the influence of sodium-glucose cotransporter-2 inhibitors on [ 18 F]-fluorodeoxyglucose-PET results.

To investigate the value (short-term and long-term outcomes) of imaging in addition to clinical monitoring versus clinical monitoring only.

To investigate the value of imaging in a treat-to-target approach in GCA and TAK.

To investigate the value of imaging for response assessment and correlation with patient-reported outcomes in GCA and TAK.

To investigate the association of imaging results with novel laboratory biomarkers in GCA and TAK.

To define remission of imaging results in GCA and TAK.

Validate cut-off values for intima–media thickness at several vessels in special patient populations such as those at advanced age or with severe arteriosclerosis.

To compare the performance of MR-angiography and CT-angiography for the evaluation of arterial stenoses and aneurysms in GCA and TAK.

To study the value of echocardiography (transthoracic and transoesophageal) for the diagnosis and follow-up of aortitis.

To investigate the optimal frequency and modality to screen for vascular damage in GCA and TAK patients; to identify predictors of vascular damage.

To determine the added value of very high-resolution ultrasound (≥50 MHz) of temporal arteries.

To calculate the number of patients needed to screen with imaging for preventing one additional aortic complication (aneurysm or dissection).

To investigate the sensitivity and specificity of imaging to detect ongoing or relapsing inflammation versus remodelling or structural damage of vessels.

To study the association between the progression of vascular damage and imaging detected inflammation, particularly whether inflammatory lesions at the aorta and other large arteries progress to structural damage once they are resolved.

To test imaging as an outcome tool in randomised controlled trials.

The EULAR recommendations for the use of imaging in LVV in clinical practice have been updated into a set of three overarching principles and eight recommendations. This update led to important changes in some statements from the original recommendations, particularly concerning the investigation of axillary arteries by ultrasound in patients with suspected GCA, the use of FDG-PET for the diagnosis of GCA and the use of imaging for the assessment of patients with GCA or TAK during follow-up. 3 As emphasised in 2018 and also in this manuscript, it is not the intention of the EULAR imaging recommendations to dismiss the role of TAB in GCA; however, when imaging is rapidly available and reveals clear signs that are in concordance with the clinical picture, the added value of performing a TAB is uncertain. In such situations, TAB would expose patients to an unnecessary, although small, risk of complications from the surgical procedure as well as to unnecessary GC treatment until histology results become available. 88 In contrast, the 2021 American College of Rheumatology/Vasculitis Foundation Guideline for the Management of GCA and TAK recommended that TAB should be the primary diagnostic test in GCA. 12 The ACR committee argued that ‘rheumatologists and radiologists in the USA are less experienced in using ultrasound to diagnose temporal artery involvement in GCA compared with their counterparts in Europe’. This raises the question whether recommendations should reflect current reality or whether they can be aspirational and be used to promote a change of clinical routine, particularly when the supporting evidence is strong. 11

The value of imaging for monitoring was one of the topics that were discussed most during the meeting. The task force recognised the need for new objective markers of inflammation in GCA and TAK, given that several new drugs directly influence acute phase reactants rendering them unreliable for the assessment of disease activity. 89 Studies investigating clinical+imaging-based monitoring in comparison to clinical monitoring alone, currently lacking, have therefore a high priority on the current research agenda. Another hot topic was the assessment of vascular damage during follow-up. While it is generally agreed that vascular damage including aortic dilatation is more common in patients with GCA than in the general population, it is less clear how many emergency surgical interventions or deaths from dissections can be prevented if regular imaging is conducted. 82 A related question is to what extent relapsing PMR with constitutional symptoms may be a reservoir for LV-GCA and unexplored vascular damage. 10 42 Other aspects that arose during the discussion were the value of imaging for treat-to-target strategies in GCA and TAK, the value of imaging for the assessment of treatment response in GCA, the role of artificial intelligence in evaluating images from vasculitis patients and the value of contrast-enhanced ultrasound for the assessment of vascular inflammation. 50 51 90–93 These aspects have been added to the research agenda which should stimulate investigators to address the numerous open questions in the field.

The voice of the patient representative (the second could not participate due to health reasons) was critical in the formulation of some recommendations. She particularly emphasised the importance of early treatment for patients with suspected LVV rather than withholding therapy until all diagnostic tests have been concluded. Also, the need for training of physicians and broad availability of imaging techniques, particularly ultrasound, were other important points emphasised by the task force.

A limitation of these recommendations is their restriction to GCA and TAK, excluding other types of LVV such as (vascular) Behçet’s disease, IgG4-related disease or LVV secondary to rheumatoid arthritis or other rheumatic diseases. Another limitation is circular reasoning in some diagnostic studies, meaning that the imaging result was used (at least in part) by the clinician to establish the final diagnosis. While this is sometimes unavoidable because of the uncertainty related to the clinical diagnosis, it might lead to an overestimation of the value of the imaging technique. We, therefore, conducted subanalyses excluding studies with circular reasoning to confirm the robustness of the primary results. 11 Virtually no new evidence has been generated for the use of imaging in TAK, consequently, the respective recommendations remained largely unchanged. 11 Despite these limitations, we believe that the updated recommendations represent a step forward towards a better diagnosis and follow-up of patients with GCA and TAK.

Implementation of these recommendations is another critical step, as also emphasised by an EULAR initiative on the implementation of recommendations. 94 We plan to present and discuss the updated recommendations at national and international conferences, and to continue promoting training courses in imaging for these diseases at a national and international level. Anchoring imaging in training curricula for rheumatologists would also be a fundamental step to ensure implementation of these recommendations in practice. In addition, we envision adaption of the recommendations to the local requirements, promoted by our task force members residing in different EULAR countries. We plan to assess possible barriers and facilitators to identify points for improvement. The lack of availability of technical equipment, specifically high-end ultrasound machines in the rheumatology outpatient clinics, might be an important barrier to implementation, and we hope that these recommendations will help to convince payors that adequate equipment is required to deliver high-quality care to patients with suspected GCA and TAK. The delay between the clinical consultation and the conduction of imaging as well as the percentage of patients with suspected LVV undergoing imaging might be important quality indicators for the level of implementation of these recommendations.

In summary, three overarching principles and eight recommendations are available to guide the use of imaging for the diagnosis and follow-up assessment of GCA and TAK in clinical practice. These recommendations are based on evidence and expert consensus. The research agenda is also an important product of this work, highlighting the gaps of knowledge and areas for further studies. The next update is expected to be undertaken when sufficient new evidence has become available on imaging in LVV. It is our vision that these recommendations standardise and optimise the use of imaging in the diagnosis and assessment of people living with GCA and TAK.

Ethics statements

Patient consent for publication.

Not applicable.

Acknowledgments

The authors would like to thank Louise Falzon for her work in the development of the literature search strategy. We also thank Lorna Neill, Luca Cimino and Fabrizio Gozzi for their help with the update of the PICO questions.

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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

Handling editor Josef S Smolen

X @sofiaramiro82, @philipp_j_bosch, @cristinadbponte, @Sarah_L_Mackie, @ertugrul_cagri, @jmolinacollada, @profbdasgupta, @annamolto

Presented at The results of this study have been presented at EULAR 2023 (LB0009).

Contributors All authors were involved in the discussion and formulation of the recommendations. CDejaco wrote the first version of the manuscript. All authors reviewed it and made extensive comments and appropriate changes to it. All authors approved the final version of the manuscript.

Funding Funding was provided by the European Alliance of Associations for Rheumatology (Project number: QoC13).

Disclaimer The views expressed in this article are those of the authors and not necessarily those of the NIHR, the NIHR Leeds Biomedical Research Centre, the National Health Service or the UK Department of Health and Social Care.

Competing interests CDejaco has received consulting/speaker’s fees from Abbvie, Eli Lilly, Janssen, Novartis, Pfizer, Roche, Galapagos, Sparrow and Sanofi; grant support from AbbVie and Novartis, all unrelated to this manuscript. He is an editorial board member of ARD. SR has received research grants and/or consultancy fees From AbbVie, Eli Lilly, Galapagos, MSD, Novartis, Pfizer, Sanofi, UCB. MB: consultancy fees from AbbVie PB has received speaker fees by Janssen and project grants by Pfizer. CP has received research grants and/or consultancy fees from AbbVie, Vifor, Roche, GlaxoSmithKline and AstraZeneca, all unrelated to this manuscript. SLM reports: Consultancy on behalf of her institution for Roche/Chugai, Sanofi, AbbVie, AstraZeneca; Investigator on clinical trials for Sanofi, GSK, Sparrow; speaking/lecturing on behalf of her institution for Roche/Chugai, Vifor, Pfizer and Novartis; chief investigator on STERLING-PMR trial, funded by NIHR; patron of the charity PMRGCAuk. No personal remuneration was received for any of the above activities. Support from Roche/Chugai to attend EULAR2019 in person and from Pfizer to attend ACR Convergence 2021 virtually. SLM is supported in part by the NIHR Leeds Biomedical Research Centre. TAB reports research grants from Deutsche Forschungsgemeinschaft (DFG) and Siemens Healthineers on behalf of his Department. He has received consulting/speaker’s fees from BioTel Research, Chugai, Guerbet, Novartis, Roche, Sanofi and Siemens Healthineers. DB consultancy fees from Roche and GSKSara Brolin: Grant from Novartis. MCC has received consulting fees from GSK, SCL-Vifor, AbbVie, AstraZeneca and Janssen, and a research grant form Kiniksa Pharmaceuticals. JM-C has received consulting/speaker’s fees from Abbvie, Lilly, Janssen, Novartis, Pfizer, UCB, MSD, all unrelated to this manuscript. BD has received consultancies and educational grants from Novartis, Abbvie, Roche, Chugai, Sanofi. BDN has received consulting/speaker’s fees from Roche and Novartis all unrelated to this manuscript. EDM Research funding/consulting and conferences fees from: Abbvie, Novartis, Pfizer, Roche, Janssen, Lilly, MSD, BMS, UCB, Grunental and Sanofi. CDuftner has received consulting/speaker’s fees from Abbvie, AOP Orphan, Astra-Zeneca, Bristol-Myers-Squibb, Eli-Lilly, Janssen, Galapagos, Merck-Sharp-Dohme, Novartis, Pfizer, Roche, Sandoz, UCB, Vifor, and grant/research support from Eli-Lilly, Pfizer, UCBHaner Direskeneli is investigator in clinical trials for Abbvie and Novartis, had educational support from Pfizer, Amgene, Celltrion, UCB and Roche unrelated to this manuscript. AM has received research grants and/or consultancy fees From AbbVie, BMS, Biogen, Eli Lilly, Galapagos, Janssen, MSD, Novartis and UCB. LS has received grant support from the Swiss Society of Rheumatology, iQone and Sandoz and support for travel expenses from Sanofi; all unrelated to this manuscript. RHJAS has received independent research grants of Siemens Healtineers and WAS has received consultancy fees, honoraria and travel expenses from Abbvie, Chugai, GlaxoSmithKline, Medac, Novartis, Roche, and Sanofi and is principal investigator in trials sponsored by Abbvie, GlaxoSmithKline, Novartis and Sanofi.

Patient and public involvement statement Patient research partners were involved in the design, conduct, reporting, and dissemination plans of this research.

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.

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    DOI: 10.7759/cureus.60409 Corpus ID: 269852321; Acute Calcific Tendinitis of the Longus Colli: A Case Report and Review of the Literature @article{Mohyeldin2024AcuteCT, title={Acute Calcific Tendinitis of the Longus Colli: A Case Report and Review of the Literature}, author={Moiud Mohyeldin and Harendra Singh and Mijjal Shrestha and Mauricio Leitao and Jai Kumar}, journal={Cureus}, year={2024 ...

  27. Literature Review as Research Methodology: Aid of Ai (Artificial

    Employing a systematic literature review and content analysis, the research scrutinizes peer-reviewed articles, reports, and case studies from 2014 to 2024, focusing on the application of AI in ...

  28. Citizen journalism in Africa: A systematic literature review

    The researchers applied a systematic literature review method to the dataset, and after applying the protocol and the analysis model, a corpus of 27 articles was obtained in June 2023, covering a ...