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VINE Journal of Information and Knowledge Management Systems

ISSN : 2059-5891

Article publication date: 8 May 2017

The purpose of this paper is to demonstrate the value of using a systematic literature review to develop an integrated framework for information and knowledge management systems.

Design/methodology/approach

First, the systematic literature review method is introduced, differentiating it from traditional literature reviews in terms of value-added and limitations. Second, this methodology is used in a research application focused on absorptive capacity internal capabilities with regard to the processes of acquisition, assimilation, transformation and exploitation. Third, an integrated framework for information and knowledge management systems is developed from this application.

The systematic literature review approach provides a rigor that can assist in reducing researcher bias while simultaneously enabling the definition of a precise scope of review, with a clear explanation of selection criteria with the objective to find and review all the studies that are relevant to the search definitions. As a research method, it effectively supports a qualitative, quantitative or mixed methodology.

Research limitations/implications

This methodology was applied to one specific area of research. Specific limitations include the availability of articles in subscribed databases and the analytical capabilities of the tools used for text mining and analytics.

Originality/value

This paper demonstrates the usefulness of the systematic literature review methodology in developing an integrated framework for analysis.

• Absorptive capacity
• Systematic literature review
• Knowledge management systems
• Information systems
• Integrated framework
• Meta-routines

Senivongse, C. , Bennet, A. and Mariano, S. (2017), "Utilizing a systematic literature review to develop an integrated framework for information and knowledge management systems", VINE Journal of Information and Knowledge Management Systems , Vol. 47 No. 2, pp. 250-264. https://doi.org/10.1108/VJIKMS-03-2017-0011

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• Research article
• Open access
• Published: 16 May 2019

An analysis of current practices in undertaking literature reviews in nursing: findings from a focused mapping review and synthesis

• Helen Aveyard   ORCID: orcid.org/0000-0001-5133-3356 1 &
• Caroline Bradbury-Jones 2  

BMC Medical Research Methodology volume  19 , Article number:  105 ( 2019 ) Cite this article

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In this paper we discuss the emergence of many different methods for doing a literature review. Referring back to the early days, when there were essentially two types of review; a Cochrane systematic review and a narrative review, we identify how the term systematic review is now widely used to describe a variety of review types and how the number of available methods for doing a literature review has increased dramatically. This led us to undertake a review of current practice of those doing a literature review and the terms used to describe them.

We undertook a focused mapping review and synthesis. Literature reviews; defined as papers with the terms review or synthesis in the title, published in five nursing journals between January 2017–June 2018 were identified. We recorded the type of review and how these were undertaken.

We identified more than 35 terms used to describe a literature review. Some terms reflected established methods for doing a review whilst others could not be traced to established methods and/or the description of method in the paper was limited. We also found inconsistency in how the terms were used.

We have identified a proliferation of terms used to describe doing a literature review; although it is not clear how many distinct methods are being used. Our review indicates a move from an era when the term narrative review was used to describe all ‘non Cochrane’ reviews; to a time of expansion when alternative systematic approaches were developed to enhance rigour of such narrative reviews; to the current situation in which these approaches have proliferated to the extent so that the academic discipline of doing a literature review has become muddled and confusing. We argue that an ‘era of consolidation’ is needed in which those undertaking reviews are explicit about the method used and ensure that their processes can be traced back to a well described, original primary source.

Peer Review reports

Over the past twenty years in nursing, literature reviews have become an increasingly popular form of synthesising evidence and information relevant to the profession. Along with this there has been a proliferation of publications regarding the processes and practicalities of reviewing [ 1 , 2 , 3 , 4 ], This increase in activity and enthusiasm for undertaking literature reviews is paralleled by the foundation of the Cochrane Collaboration in 1993. Developed in response to the need for up-to-date reviews of evidence of the effectiveness of health care interventions, the Cochrane Collaboration introduced a rigorous method of searching, appraisal and analysis in the form of a ‘handbook’ for doing a systematic review [ 5 ] .Subsequently, similar procedural guidance has been produced, for example by the Centre for Reviews and Dissemination (CRD) [ 6 ] and The Joanna Briggs Institute [ 7 ]. Further guidance has been published to assist researchers with clarity in the reporting of published reviews [ 8 ].

In the early days of the literature review era, the methodological toolkit for those undertaking a literature was polarised, in a way that mirrored the paradigm wars of the time within mixed-methods research [ 9 ]. We refer to this as the ‘dichotomy era’ (i.e. the 1990s), The prominent methods of literature reviewing fell into one of two camps: The highly rigorous and systematic, mostly quantitative ‘Cochrane style’ review on one hand and a ‘narrative style’ review on the other hand, whereby a body of literature was summarised qualitatively, but the methods were often not articulated. Narrative reviews were particularly popular in dissertations and other student work (and they continue to be so in many cases) but have been criticised for a lack of systematic approach and consequently significant potential for bias in the findings [ 10 , 11 ].

The latter 1990s and early 2000, saw the emergence of other forms of review, developed as a response to the Cochrane/Narrative dichotomy. These alternative approaches to the Cochrane review provided researchers with reference points for performing reviews that drew on different study types, not just randomised controlled trials. They promoted a systematic and robust approach for all reviews, not just those concerned with effectiveness of interventions and treatments. One of the first published description of methods was Noblet and Hare’s (1998) ‘Meta-ethnography’ [ 12 ]. This method, although its name suggests otherwise, could incorporate and synthesise all types of qualitative research, not just ethnographies. The potential confusion regarding the inclusion of studies that were not ethnographies within a meta-ethnography, promoted the description of other similar methods, for example, the meta-synthesis of Walsh and Downe (2005) [ 13 ] and the thematic synthesis of Thomas and Harden (2008) [ 14 ]. Also, to overcome the dichotomy of the quantitative/qualitative reviews, the integrative review was described according to Whitemore and Knafl (2005) [ 15 ]. These reviews can be considered to be literature reviews that have been done in a systematic way but not necessarily adhering to guidelines established by the Cochrane Collaboration. We conceptualise this as the ‘expansion era’. Some of the methods are summarised in Table  1 .

Over the past two decades there has been a proliferation of review types, with corresponding explosion of terms used to describe them. A review of evidence synthesis methodologies by Grant and Booth in 2009 [ 20 ] identified 14 different approaches to reviewing the literature and similarly, Booth and colleagues [ 21 ] detailed 19 different review types, highlighting the range of review types currently available. We might consider this the ‘proliferation era’. This is however, somewhat a double-edged sword, because although researchers now have far more review methods at their disposal, there is risk of confusion in the field. As Sabatino and colleagues (2014) [ 22 ] have argued, review methods are not always consistently applied by researchers.

Aware of such potential inconsistency and also our own confusion at times regarding the range of review methods available, we questioned what was happening within our own discipline of nursing. We undertook a snap-shot, contemporary analysis to explore the range of terms used to describe reviews, the methods currently described in nursing and the underlying trends and patterns in searching, appraisal and analysis adopted by those doing a literature review. The aim was to gain some clarity on what is happening within the field, in order to understand, explain and critique what is happening within the proliferation era.

In order to explore current practices in doing a literature review, we undertook a ‘Focused Mapping Review and Synthesis’ (FMRS) – an approach that has been described only recently. This form of review [ 19 ] is a method of investigating trends in academic publications and has been used in a range of issues relevant to nursing and healthcare, for example, theory in qualitative research [ 23 ] and vicarious trauma in child protection research [ 24 ].

A FMRS seeks to identify what is happening within a particular subject or field of inquiry; hence the search is restricted to a particular time period and to pre-identified journals. The review has four distinct features: It: 1) focuses on identifying trends in an area rather than a body of evidence; 2) creates a descriptive map or topography of key features of research within the field rather than a synthesis of findings; 3) comments on the overall approach to knowledge production rather than the state of the evidence; 4) examines this within a broader epistemological context. These are translated into three specific focused activities: 1) targeted journals; 2) a specific subject; 3) a defined time period. The FMRS therefore, is distinct from other forms of review because it responds to questions concerned with ‘what is happening in this field?’ It was thus an ideal method to investigate current practices in literature reviews in nursing.

Using the international Scopus (2016) SCImago Journal and Country Rank, we identified the five highest ranked journals in nursing at that time of undertaking the review. There was no defined method for determining the number of journals to include in a review; the aim was to identify a sample and we identified five journals in order to search from a range of high ranking journals. We discuss the limitations of this later. Journals had to have ‘nursing’ or ‘nurse’ in the title and we did not include journals with a specialist focus, such as nutrition, cancer etcetera. The included journals are shown in Table  2 and are in order according to their ranking. We recognise that our journal choice meant that only articles published in English made it into the review.

A key decision in a FMRS is the time-period within which to retrieve relevant articles. Like many other forms of review, we undertook an initial scoping to determine the feasibility and parameters of the project [ 19 ]. In our previous reviews, the timeframe has varied from three months [ 23 ] to 6 years [ 24 ]. The main criterion is the likelihood for the timespan to contain sufficient articles to answer the review questions. We set the time parameter from January 2017–June 2018. We each took responsibility for two and three journals each from which to retrieve articles. We reviewed the content page of each issue of each journal. For our purposes, in order to reflect the diverse range of terms for describing a literature review, as described earlier in this paper, any paper that contained the term ‘review’ or ‘synthesis’ in the title was included in the review. This was done by each author individually but to enhance rigour, we worked in pairs to check each other’s retrieval processes to confirm inter-rater consistency. This process allowed any areas of uncertainty to be discussed and agreed and we found this form of calibration crucial to the process. The inclusion and exclusion criteria are shown in Table  3 .

Articles meeting the inclusion criteria, papers were read in full and data was extracted and recorded as per the proforma developed for the study (Table 4 ). The proforma was piloted on two papers to check for usability prior to data extraction. Data extraction was done independently but we discussed a selection of papers to enhance rigour of the process. No computer software was used in the analysis of the data. We did not critically appraise the included studies for quality because our purpose was to profile what is happening in the field rather than to draw conclusions from the included studies’ findings.

Once the details from all the papers had been extracted onto the tables, we undertook an analysis to identify common themes in the included articles. Because our aim was to produce a snap-shot profile, our analysis was thematic and conceptual. Although we undertook some tabulation and numerical analysis, our primary focus was on capturing patterns and trends characterised by the proliferation era. In line with the FMRS method, in the findings section we have used illustrative examples from the included articles that reflect and demonstrate the point or claim being made. These serve as useful sources of information and reference for readers seeking concrete examples.

Between January 2017 and June 2018 in the five journals we surveyed, a total of 222 papers with either ‘review’ or ‘synthesis’ in the title were retrieved and included in our analysis. We identified three primary themes: 1) Proliferation in names for doing a review; 2) Allegiance to an established review method; 3) Clarity about review processes. The results section is organised around these themes.

Proliferation in names for doing a review

We identified more than 35 terms used by authors to describe a literature review. Because we amalgamated terms such as ‘qualitative literature review’ and ‘qualitative review’ the exact number is actually slightly higher. It was clear from reading the reviews that many different terms were used to describe the same processes. For example qualitative systematic review, qualitative review and meta-synthesis, qualitative meta-synthesis, meta-ethnography all refer to a systematic review of qualitative studies. We have therefore grouped together the review types that refer to a particular type of review as described by the authors of the publications used in this study (Table  5 ).

In many reviews, the specific type of review was indicated in the title as seen for example in Table  5 . A striking feature was that all but two of the systematic reviews that contained a meta-analysis were labelled as such in the title; providing clarity and ease of retrieval. Where a literature review did not contain a meta-analysis, the title of the paper was typically referred to a ‘systematic review’; the implication being that a systematic review is not necessarily synonymous with a meta-analysis. However as discussed in the following section, this introduced some muddying of water, with different interpretations of what systematic review means and how broadly this term is applied. Some authors used the methodological type of included papers to describe their review. For example, a Cochrane-style systematic review was undertaken [ 25 ] but the reviewers did not undertake a meta-analysis and thus referred to their review as a ‘quantitative systematic review’.

Allegiance to an established literature review method

Many literature reviews demonstrated allegiance to a defined method and this was clearly and consistently described by the authors. For example, one team of reviewers [ 26 ] articulately described the process of a ‘meta-ethnography’ and gave a detailed description of their study and reference to the origins of the method by Noblet and Hare (1988) [ 12 ]. Another popular method was the ‘integrative review’ where most authors referred to the work of one or two seminal papers where the method was originally described (for example, Whitemore & Knafl 2005 [ 15 ]).

For many authors the term systematic review was used to mean a review of quantitative research, but some authors [ 27 , 28 , 29 ],used the term systematic review to describe reviews containing both qualitative and quantitative data.

However in many reviews, commitment to a method for doing a literature review appeared superficial, undeveloped and at times muddled. For example, three reviews [ 30 , 31 , 32 ] , indicate an integrative review in the title of their review, but this is the only reference to the method; there is no further reference to how the components of an integrative review are addressed within the paper. Other authors do not state allegiance to any particular method except to state a ‘literature review’ [ 33 ] but without an outline of a particular method for doing so. Anther review [ 34 ] reports a ‘narrative review’ but does not give further information about how this was done, possibly indicative of the lack of methods associated with the traditional narrative review. Three other reviewers documented how they searched, appraised and analysed their literature but do not reference an over-riding approach for their review [ 35 , 36 , 37 ]. In these examples, the review can be assumed to be a literature review, but the exact approach is not clear.

In other reviews, the methods for doing a literature review appear to be used interchangeably. For example in one review [ 38 ] the term narrative review was used in the title but in the main text an integrative review was described. In another review [ 39 ] two different and distinct methods were combined in a ‘meta-ethnographic meta-synthesis’.

Some authors [ 40 , 41 ] referred to a method used to undertake their review, for example a systematic review, but did not reference the primary source from where the method originated. Instead a secondary source, such as a textbook is used to reference the approach taken [ 20 , 42 ].

Clarity about review processes

Under this theme we discerned two principal issues: searching and appraisal. The majority of literature reviews contain three components- searching, appraisal and analysis, details of which are usually reported in the methods section of the papers. However, this is not always the case and for example, one review [ 43 ] provides only a search strategy with no information about the overall method or how critical appraisal or analysis were undertaken. Despite the importance of the process of analysis, we found little discussion of this in the papers reviewed.

The overwhelming trend for those doing a literature review was to describe a comprehensive search; although for many in our sample, a comprehensive search appeared to be limited to a database search; authors did not describe additional search strategies that would enable them to find studies that might be missed through electronic searching. Furthermore, authors did not define what a comprehensive search entailed, for example whether this included grey literature. We identified a very small number of studies where authors had undertaken a purposive sample [ 26 , 44 ]; in these reviews authors clearly stated that their search was for ‘seminal papers’ rather than all papers.

We reviewed the approaches to critical appraisal described in the papers and there were varying interpretations of what this means and which aspect of the included articles were to be subject to appraisal. Some authors [ 36 , 45 , 46 ] used the term ‘critical appraisal’ to refer to relevance of the paper to the review, rather than quality criteria. In that sense critical appraisal was used more as an inclusion criterion regarding relevance, rather than quality in the methods used. Mostly though, the term was used to describe the process of critical analysis of the methodological quality of included papers included in a review. When the term was used in this way to refer to quality criteria, appraisal tools were often used; for example, one review [ 47 ] provides a helpful example when they explain how a particular critical appraisal tool was used to asses the quality of papers in their review. Formal critical appraisal was undertaken by the vast majority reviewers, however the role of critical appraisal in the paper was often not explained [ 33 , 48 ]. It was common for a lot of detail to be provided about the approach to appraisal, including how papers were assessed and how disagreements between reviewers about the quality of individual papers were resolved, with no further mention of the subsequent role of the appraisal in the review. The reason for doing the critical appraisal in the review was often unclear and furthermore, in many cases, researchers included all papers within their review regardless of quality. For example, one team of reviewers [ 49 ] explained how the process, in their view, is not to exclude studies but to highlight the quality of evidence available. Another team of reviewers described how they did not exclude studies on the basis of quality because of the limited amount of research available on the topic [ 50 ].

Our review has identified a multiplicity of similar terms and approaches used by authors when doing a literature review, that we suggests marks the ‘proliferation era’. The expansion of terms used to describe a literature review has been observed previously [ 19 , 21 ]. We have identified an even wider range of terms, indicating that this trend may be increasing. This is likely to give the impression of an incoherent and potentially confusing approach to the scholarly undertaking of doing a literature review and is likely to be particularly problematic for novice researchers and students when attempting to grapple with the array of approaches available to them. The range of terms used in the title of papers to describe a literature review may cause both those new to research to wonder what the difference is between a qualitative evidence synthesis and a qualitative systematic review and which method is most suitable for their enquiry.

The clearest articles in our review were those that reported a systematic review with or without a meta-analysis. For example, one team of reviewers [ 25 ] undertook a Cochrane-style systematic review but did not undertake a meta-analysis and thus referred to their review as a ‘quantitative systematic review’. We found this form of labelling clear and helpful and is indeed in line with current recommendations [ 8 ]. While guidelines exist for the publication of systematic reviews [ 8 , 51 ], given the range of terms that are used by authors, some may be unclear when these guidelines should apply and this adds some confusion to the field. Of course, authors are at liberty to call their review processes whatever they deem appropriate, but our analysis has unearthed some inconsistencies that are confusing to the field of literature reviewing.

There is current debate about the status of literature reviews that are not ‘Cochrane’ style reviews [ 52 ]. Classification can be complex and whilst it might be tempting to refer to all non Cochrane-style reviews as ‘narrative reviews’ [ 52 ], literature reviews that conform to a recognised method would generally not be considered as such [ 53 ] and indeed the Cochrane Collaboration handbook refers to the principles of systematic review as applicable to different types of evidence, not just randomised controlled trials [ 5 ] .This raises the question as to whether the term systematic review should be an umbrella term referring to any review with an explicit method; which is implicit in the definition of a systematic review, but which raises the question as to how rigorous a method has to be to meet these standards, a thorny issue which we have identified in this study.

This review has identified a lack of detail in the reporting of the methods used by those doing a review. In 2017, Thorne raised the rhetorical question: ‘What kind of monster have we created?‘ [ 54 ]. Critiquing the growing investment in qualitative metasyntheses, she observed that many reviews were being undertaken that position themselves as qualitative metasyntheses, yet are theoretically and methodologically superficial. Thorne called for greater clarity and sense of purpose as the ‘trend in synthesis research marches forward’ [ 54 ]. Our review covered many review types, not just the qualitative meta-synthesis and its derivatives. However, we concur with Thorne’s conclusion that research methods are not extensively covered or debated in many of the published papers which might explain the confusion of terms and mixing of methods.

Despite the proliferation in terms for doing a literature review, and corresponding associated different methods and a lack of consistency in their application, our review has identified how the methods used (or indeed the reporting of the methods) appear to be remarkably similar in most publications. This may be due to limitations in the word count available to authors. However for example, the vast majority of papers describe a comprehensive search, critical appraisal and analysis. The approach to searching is of particular note; whilst comprehensive searching is the cornerstone of the Cochrane approach, other aproaches advocate that a sample of literature is sufficient [ 15 , 20 ]. Yet in our review we found only two examples where reviewers had used this approach, despite many other reviews claiming to be undertaking a meta-ethnography or meta-synthesis. This indicates that many of those doing a literature review have defaulted to the ‘comprehensive search’ irrespective of the approach to searching suggested in any particular method which is again indicative of confusion in the field.

Differences are reported in the approach to searching and critical appraisal and these appear not to be linked to different methods, but seem to be undertaken on the judgement and discretion of the reviewers without rationale or justification within the published paper. It is not for us to question researchers’ decisions as regards managing the flow of articles through their reviews, but when it comes to the issue of both searching and lack of clarity about the role of critical appraisal there is evidence of inconsistency by those doing a literature review. This reflects current observations in the literature where the lack of clarity about the role of critical appraisal within a literature review is debated . [ 55 , 56 ].

Our review indicates that many researchers follow a very similar process, regardless of their chosen method and the real differences that do exist between published methods are not apparent in many of the published reviews. This concurs with previously mentioned concerns [ 54 ] about the superficial manner in which methods are explored within literature reviews. The overriding tendency is to undertake a comprehensive review, critical appraisal and analysis, following the formula prescribed by Cochrane, even if this is not required by the literature review method stated in the paper. Other researchers [ 52 ] have questioned whether the dominance of the Cochrane review should be questioned. We argue that emergence of different methods for doing a literature review in a systematic way has indeed challenged the perceived dominance of the Cochrane approach that characterised the dichotomy era, where the only alternative was a less rigourous and often poorly described process of dealing with literature. It is positive that there is widespread acknowledgement of the validity of other approaches. But we argue that the expansion era, whereby robust processes were put forward as alternatives that filled the gap left by polarisation, has gone too far. The magnitude in the number of different approaches identified in this review has led to a confused field. Thorne [ 54 ] refers to a ‘meta-madness’; with the proliferation of methods leading to the oversimplification of complex literature and ideas. We would extend this to describe a ‘meta-muddle’ in which, not only are the methods and results oversimplified, but the existence of so many terms used to describe a literature review, many of them used interchangeably, has added a confusion to the field and prevented the in-depth exploration and development of specific methods. Table  6 shows the issues associated with the proliferation era and importantly, it also highlights the recommendations that might lead to a more coherent reviewing community in nursing.

The terms used for doing a literature review are often used both interchangeably and inconsistently, with minimal description of the methods undertaken. It is not surprising therefore that some journal editors do not index these consistently within the journal. For example, in one edition of one journal included in the review, there are two published integrative reviews. One is indexed in the section entitled as a ‘systematic review’, while the other is indexed in a separate section entitled ‘literature review’. In another edition of a journal, two systematic reviews with meta-analysis are published. One is listed as a research article and the other as a review and discussion paper. It seems to us then, that editors and publishers might sometimes also be confused and bewildered themselves.

Whilst guidance does exist for the publication of some types of systematic reviews in academic journals; for example the PRISMA statement [ 8 ] and Entreq guidelines [ 51 ], which are specific to particular qualitative synthesis, guidelines do not exist for each approach. As a result, for those doing an alternative approach to their literature review, for example an integrative review [ 15 ], there is only general publication guidance to assist. In the current reviewing environment, there are so many terms, that more specific guidance would be impractical anyway. However, greater clarity about the methods used and halting the introduction of different terms to mean the same thing will be helpful.

Limitations

This study provides a snapshot of the way in which literature reviews have been described within a short publication timeframe. We were limited for practical reasons to a small section of high impact journals. Including a wider range of journals would have enhanced the transferability of the findings. Our discussion is, of course, limited to the review types that were published in the timeframe, in the identified journals and which had the term ‘review’ or ‘synthesis’ in the title. This would have excluded papers that were entitled ‘meta-analysis’. However as we were interested in the range of reviews that fall outside the scope of a meta-analysis, we did not consider that this limited the scope of the paper. Our review is further limited by the lack of detail of the methods undertaken provided in many of the papers reviewed which, although providing evidence for our arguments, also meant that we had to assume meaning that was unclear from the text provided.

The development of rigorous methods for doing a literature review is to be welcomed; not all review questions can be answered by Cochrane style reviews and robust methods are needed to answer review questions of all types. Therefore whilst we welcome the expansion in methods for doing a literature review, the proliferation in the number of named approaches should be, in our view, a cause for reflection. The increase in methods could be indicative of an emerging variation in possible approaches; alternatively, the increase could be due to a lack of conceptual clarity where, on closer inspection, the methods do not differ greatly and could indeed be merged. Further scrutiny of the methods described within many papers support the latter situation but we would welcome further discussion about this. Meanwhile, we urge researchers to make careful consideration of the method they adopt for doing a literature review, to justify this approach carefully and to adhere closely to its method. Having witnessed an era of dichotomy, expansion and proliferation of methods for doing a literature review, we now seek a new era of consolidation.

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Aveyard, H., Bradbury-Jones, C. An analysis of current practices in undertaking literature reviews in nursing: findings from a focused mapping review and synthesis. BMC Med Res Methodol 19 , 105 (2019). https://doi.org/10.1186/s12874-019-0751-7

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The scope and impact of mobile health clinics in the United States: a literature review

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• 1 Li Ka Shing Faculty of Medicine, The University of Hong Kong, 21 Sassoon Rd, Pokfulam, Hong Kong, Special Administrative Region of China. [email protected].
• 2 Department of Global Health and Social Medicine, Harvard Medical School, c/o The Family Van, 1542 Tremont St, Roxbury, MA, 02120, USA.
• 3 Harvard Business School, Soldiers Field, Boston, MA, 02163, USA.
• 4 Harvard University T.H. Chan School of Public Health, 677 Huntington Ave, Boston, MA, 02115, USA.
• 5 The Family Van: Harvard Medical School, 1542 Tremont St, Roxbury, MA, 02120, USA.
• 6 Harvard Medical School, 260 Longwood Ave, Suite 244, Boston, MA, 02115, USA.
• 7 Department of Anesthesia and Critical Care, Beth Israel Deaconess Medical Center, 330 Brookline Ave, Boston, MA, 02215, USA.
• PMID: 28982362
• PMCID: PMC5629787
• DOI: 10.1186/s12939-017-0671-2

As the U.S. healthcare system transforms its care delivery model to increase healthcare accessibility and improve health outcomes, it is undergoing changes in the context of ever-increasing chronic disease burdens and healthcare costs. Many illnesses disproportionately affect certain populations, due to disparities in healthcare access and social determinants of health. These disparities represent a key area to target in order to better our nation's overall health and decrease healthcare expenditures. It is thus imperative for policymakers and health professionals to develop innovative interventions that sustainably manage chronic diseases, promote preventative health, and improve outcomes among communities disenfranchised from traditional healthcare as well as among the general population. This article examines the available literature on Mobile Health Clinics (MHCs) and the role that they currently play in the U.S. healthcare system. Based on a search in the PubMed database and data from the online collaborative research network of mobile clinics MobileHealthMap.org , the authors evaluated 51 articles with evidence on the strengths and weaknesses of the mobile health sector in the United States. Current literature supports that MHCs are successful in reaching vulnerable populations, by delivering services directly at the curbside in communities of need and flexibly adapting their services based on the changing needs of the target community. As a link between clinical and community settings, MHCs address both medical and social determinants of health, tackling health issues on a community-wide level. Furthermore, evidence suggest that MHCs produce significant cost savings and represent a cost-effective care delivery model that improves health outcomes in underserved groups. Even though MHCs can fulfill many goals and mandates in alignment with our national priorities and have the potential to help combat some of the largest healthcare challenges of this era, there are limitations and challenges to this healthcare delivery model that must be addressed and overcome before they can be more broadly integrated into our healthcare system.

Keywords: Chronic disease management; Community-clinical linkage; Cost-effectiveness; Health disparities; Mobile health clinics; Population health; Preventative care; Social determinants of health.

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• Chronic Disease / prevention & control
• Delivery of Health Care / economics
• Delivery of Health Care / organization & administration
• Health Services Accessibility
• Health Services Research*
• Mobile Health Units*
• United States

Bibliometric Review Methodology and State of the Science Bibliometric Review of Research on Problem-based Learning, 2017-2021

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Bibliometric reviews of research have gained increased popularity since the mid-2010s. Yet, many readers may be uncertain as to the purpose and method of bibliometric reviews. This article provides an introduction to bibliometric review methods, clarifying their purpose, as well as methods of data collection, analysis, and interpretation. The latter portion of the article is devoted to a state-of-the-science, bibliometric review of research on problem-based learning. In this review, the author applied descriptive statistics, citation analysis, and co-citation analysis to a database of 5,764 Scopus-indexed documents on problem-based learning published between 2017 and 2021. The analyses found increasing diversity of the PBL knowledge base with respect to both geographical, subject area origins, and focus of the research when compared with findings reported in past reviews. Author co-citation analysis visualized the intellectual structure of this recent PBL literature revealing four dominant schools of thought: Interdisciplinary PBL Theory and Practice, Active Learning, Social and Experiential Learning, PBL Process. This finding highlights the increasing integration of PBL with other methods of experiential and active learning.

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

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

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

The Literature Review Defined

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

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

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

Purpose and Importance of the Literature Review

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

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

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

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

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

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

Approaching the Literature Review

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

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

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

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

Locating and Organizing the Literature

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

Human Resources

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

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

Strategies for Finding Related Researcher Publications in Databases and Search Engines

Search Tools and Related Literature

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

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

Box Information Resources

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

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

Getting Organized

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

Citation Managers

Knowing When to Say When

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

Putting It All Together

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

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

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

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

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Toward a framework for selecting indicators of measuring sustainability and circular economy in the agri-food sector: a systematic literature review

• LIFE CYCLE SUSTAINABILITY ASSESSMENT
• Published: 02 March 2022

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• Cecilia Silvestri   ORCID: orcid.org/0000-0003-2528-601X 1 ,
• Luca Silvestri   ORCID: orcid.org/0000-0002-6754-899X 2 ,
• Michela Piccarozzi   ORCID: orcid.org/0000-0001-9717-9462 1 &
• Alessandro Ruggieri 1  

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A Correction to this article was published on 24 March 2022

This article has been updated

The implementation of sustainability and circular economy (CE) models in agri-food production can promote resource efficiency, reduce environmental burdens, and ensure improved and socially responsible systems. In this context, indicators for the measurement of sustainability play a crucial role. Indicators can measure CE strategies aimed to preserve functions, products, components, materials, or embodied energy. Although there is broad literature describing sustainability and CE indicators, no study offers such a comprehensive framework of indicators for measuring sustainability and CE in the agri-food sector.

Starting from this central research gap, a systematic literature review has been developed to measure the sustainability in the agri-food sector and, based on these findings, to understand how indicators are used and for which specific purposes.

The analysis of the results allowed us to classify the sample of articles in three main clusters (“Assessment-LCA,” “Best practice,” and “Decision-making”) and has shown increasing attention to the three pillars of sustainability (triple bottom line). In this context, an integrated approach of indicators (environmental, social, and economic) offers the best solution to ensure an easier transition to sustainability.

Conclusions

The sample analysis facilitated the identification of new categories of impact that deserve attention, such as the cooperation among stakeholders in the supply chain and eco-innovation.

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Source: Authors’ elaboration. Notes: The graph shows the temporal distribution of the articles under analysis

Source: Authors’ elaborations. Notes: The graph shows the time distribution of articles from the three major journals

Source: Authors’ elaboration. Notes: The graph shows the composition of the sample according to the three clusters identified by the analysis

Source: Authors’ elaboration. Notes: The graph shows the distribution of articles over time by cluster

Source: Authors’ elaboration. Notes: The graph shows the network visualization

Source: Authors’ elaboration. Notes: The graph shows the overlay visualization

Source: Authors’ elaboration. Notes: The graph shows the classification of articles by scientific field

Source: Authors’ elaboration. Notes: Article classification based on their cluster to which they belong and scientific field

Source: Authors’ elaboration

Source: Authors’ elaboration. Notes: The graph shows the distribution of items over time based on TBL

Source: Authors’ elaboration. Notes: The graph shows the Pareto diagram highlighting the most used indicators in literature for measuring sustainability in the agri-food sector

Source: Authors’ elaboration. Notes: The graph shows the distribution over time of articles divided into conceptual and empirical

Source: Authors’ elaboration. Notes: The graph shows the classification of articles, divided into conceptual and empirical, in-depth analysis

Source: Authors’ elaboration. Notes: The graph shows the geographical distribution of the authors

Source: Authors’ elaboration. Notes: The graph shows the distribution of authors according to the continent from which they originate

Source: Authors’ elaboration. Notes: The graph shows the time distribution of publication of authors according to the continent from which they originate

Source: Authors’ elaboration. Notes: Sustainability measurement indicators and impact categories of LCA, S-LCA, and LCC tools should be integrated in order to provide stakeholders with best practices as guidelines and tools to support both decision-making and measurement, according to the circular economy approach

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Change history, 24 march 2022.

A Correction to this paper has been published: https://doi.org/10.1007/s11367-022-02038-9

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Silvestri, C., Silvestri, L., Piccarozzi, M. et al. Toward a framework for selecting indicators of measuring sustainability and circular economy in the agri-food sector: a systematic literature review. Int J Life Cycle Assess (2022). https://doi.org/10.1007/s11367-022-02032-1

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CASE REPORT article

Antiphospholipid antibody-related hepatic vasculitis in a juvenile after non-severe covid-19: a case report and literature review.

• 1 Tsinghua Medicine, School of Medicine, Tsinghua University, Beijing, China
• 2 Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Peking Union Medical College, Beijing, China
• 3 State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Beijing, China
• 4 Department of Infectious Disease, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Peking Union Medical College, Beijing, China

Antiphospholipid antibodies (aPL) are both laboratory evidence and causative factors for a broad spectrum of clinical manifestations of antiphospholipid syndrome (APS), with thrombotic and obstetric events being the most prevalent. Despite the aPL-triggered vasculopathy nature of APS, vasculitic-like manifestations rarely exist in APS and mainly appear associated with other concurrent connective tissue diseases like systemic lupus erythematous. Several studies have characterized pulmonary capillaritis related to pathogenic aPL, suggesting vasculitis as a potential associated non-thrombotic manifestation. Here, we describe a 15-year-old girl who develops hepatic infarction in the presence of highly positive aPL, temporally related to prior non-severe COVID-19 infection. aPL-related hepatic vasculitis, which has not been reported before, contributes to liver ischemic necrosis. Immunosuppression therapy brings about favorable outcomes. Our case together with retrieved literature provides supportive evidence for aPL-related vasculitis, extending the spectrum of vascular changes raised by pathogenic aPL. Differentiation between thrombotic and vasculitic forms of vascular lesions is essential for appropriate therapeutic decision to include additional immunosuppression therapy. We also perform a systematic review to characterize the prevalence and clinical features of new-onset APS and APS relapses after COVID-19 for the first time, indicating the pathogenicity of aPL in a subset of COVID-19 patients.

1 Introduction

Antiphospholipid syndrome (APS) is a systemic autoimmune disorder characteristic of arterial, venous, or microvascular thrombosis, obstetric morbidity, and well-defined non-thrombotic manifestations in the setting of persistent antiphospholipid antibodies (aPL) ( 1 , 2 ). aPL, composed of a diverse family of acquired autoantibodies, are recognized as causative factors for clinical manifestations of APS ( 3 ). Both genetic and environmental elements could exert as precipitating factors for aPL production, with infection being the most prevalent trigger ( 4 – 6 ). In the recent COVID-19 pandemic, the observed high prevalence of aPL has been reported, yet the potential pathogenicity of these antibodies remains uncertain and controversial ( 7 ). The molecular mimicry between severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) viral proteins and native tissues and the neoepitope caused by SARS-CoV-2-induced oxidative stress probably contribute to aPL generation ( 8 ). In addition to widely reported aPL-related thrombosis, associated non-thrombotic manifestations are emerging with considerable evidence ( 9 ). Compared with adult patients, aPL-related non-thrombotic complications, both criteria and non-criteria, are more frequently presented in pediatric patients ( 10 ). aPL-related vasculitis is characterized as the inflammation of vessel walls and is only well-confirmed in pulmonary capillaries as diffuse alveolar hemorrhage (DAH) ( 11 ). This rare manifestation can result in occlusion of vessel lumen in the absence of thrombus, which might make it clinically indistinguishable to thrombotic events. Differentiation between thrombotic and vasculitic causes of aPL-related vascular damage is essential for proper therapeutic decision to adequately include immunosuppression therapy ( 12 ). Here, we describe an uncommon case of a young girl with aPL-related vasculitis-induced liver infarction after non-severe COVID-19 infection, providing valuable information for development of pathogenic aPL in infectious diseases and aPL-related vasculitic manifestations.

2 Case report

A 15-year-old girl presented to the emergency department with hyperpyrexia and abdominal pain persisting for over a month. Initial laboratory results revealed elevated inflammation markers, liver dysfunction, and prolonged activated partial thromboplastin time (aPTT) (62.2 s; normal: 25–37 s) and prothrombin time (PT) (17.8 s; normal: 11–14 s). There were no bleeding signs clinically. Abdominal CT demonstrated liver “abscess-like” lesions ( Figures 1A, B ) as well as possible cholecystitis ( Figures 1C, D ), and the histopathological examination of liver biopsy specimens confirmed the acute hepatic necrosis. An empiric anti-infective therapy was initiated with intravenous ertapenem and changed to meropenem and metronidazole later. Vitamin K and plasma transfusion were applied for correction of coagulation disorders but turned out to be ineffective. Possible pathogens were under intense exploration, but all proved negative after following tests: traditional microbiologic culture and metagenomic next-generation sequencing of peripheral blood samples and liver biopsy specimens; serology screenings for fungi, SASR-CoV-2, hepatitis viruses, TORCH pathogens, Leishmania, and mycobacteria tuberculosis; Epstein–Barr virus DNA analysis; and microscopic examination of parasites in stool samples. In addition, tumor marker analysis, bone marrow examination, specific staining of liver biopsy specimens, and ceruloplasmin test showed no abnormalities. The efficacy of anti-infective therapy was undetermined with fluctuating inflammation markers and unrelieved abdominal pain. Elevated D-dimer (14.87 mg/L; normal: 0–0.55 mg/L), fibrin and fibrinogen degradation products (FDP) (27.6 μg/mL; normal: 0–5 μg/mL), and fibrinogen (7.52 g/mL; normal: 1.8–3.5 g/mL) were also indicated. Repeated CT demonstrated enlargement or reduction of some liver lesions, as well as the emergence of new lesions. The antibiotics were improved to intravenous ertapenem and vancomycin after the re-elevation of C-reactive protein. Unexpectedly, her symptoms worsened with a re-elevated fever peak and persistent coagulation disorders ( Figure 2 ).

Figure 1 (A, B) CT scan without and with contrast demonstrates liver "abscess-like" lesions. (C, D) CT scan without and with contrast demonstrates possible cholecystitis (red arrowhead). (E, F) Histopathological examination reveals vasculitis of hepatic arteries and resultant liver infarction (black arrowhead) (bar is 50 μm).

Figure 2 Positive correlation between clinical course and the administration of dexamethasone but not antibiotics. Laboratory reference range for indicators: CRP<3.0 mg/L; ALT: 9–50 U/L. CRP, C-reactive protein; ALT, alanine aminotransferase; DXM, dextromethorphan; ETP, ertapenem; MEM, meropenem; MTR, metronidazole; VAN, vancomycin; CMZ, cefmetazole; PDN, prednisone; MMF, mycophenolate mofetil; HCQ, hydroxychloroquine.

The young girl had a medical history of mild SARS-CoV-2 Omicron variant infection, presented with only nasal congestion and fatigue. The onset of abdominal pain occurred 7 days after testing negative for SARS-CoV-2 antigen and complete remission of COVID-19 symptoms. As the efficacy of low-dose dexamethasone given during plasma transfusion could not be excluded for transient improvement of the patient ( Figure 2 ), an immune dysregulation secondary to infection was considered. Serologic testing of antinuclear antibodies and autoimmune hepatitis antibodies only revealed the presence of low titers of antinuclear antibody (1:80) and smooth muscle antibody (1:80). Autoimmune liver diseases were ruled out. Antibodies associated with other systemic autoimmune diseases were comprehensively evaluated, and the presence of IgG anticardiolipin (aCL) and IgG anti-β2-glycoprotein I (anti-β2GPI) antibodies, as well as lupus anticoagulant (LA), was demonstrated ( Table 1 ). Other autoantibodies were undetected. The aPTT and PT correction tests showed negative results. Consecutive examinations demonstrated disease progression following changes of aPL titers, and hepatic lesions were considered to be related to aPL. Considering the hypercoagulable state of pathogenic aPL, the patient underwent CT scan, MRI, MRA, and vascular ultrasound and multiple small acute to subacute cerebral infarcts were indicated. No thrombus and other abnormalities were detected. The liver biopsy specimen was histologically re-evaluated. Immune infiltration with a large number of neutrophils and fibrinoid necrosis were evident in some small- and medium-sized arteries, and adjacent hepatic tissues underwent ischemic infarction consequently ( Figures 1D–F ). The patient traversed the most severe phase because of administered low-dose dexamethasone. Further therapy included 40 mg prednisone once a day, 500 mg mycophenolate mofetil (MMF) twice a day, and 200 mg hydroxychloroquine twice a day for immunosuppression, along with 100 mg aspirin once a day to prevent future thrombosis. The patient were discharged from the hospital with reduced inflammation markers, diminished abdominal pain, and healed liver lesions as shown in CT examination. The administration of MMF, hydroxychloroquine and aspirin remained unchanged after discharge, whereas prednisone was gradually tapered. The dose reduction proceeded at 5 mg per week until reaching a daily dose of 20 mg, followed by a weekly reduction of 2.5 mg until reaching a daily dose of 15 mg. Scheduled follow-up appointments were conducted. Six months after discharge, the patient discontinued medication autonomously and subsequently experienced a relapsed right-upper quadrant pain with re-elevated aPL titers and significantly prolonged aPTT ( Table 1 ). D-dimer, FDP, and fibrinogen were within normal ranges. Resumption of treatment yielded amelioration. Considering the persistence of medium-to-highly positive aCL and LA for over 12 weeks, as well as aPL-related hepatic vasculitis and cerebral infarction, the diagnosis was made as highly probable APS with vasculitis as a non-criteria manifestation.

Table 1 Disease parameters and titers of autoantibodies.

3 Discussion

Compared with the updated Sapporo criteria with only vascular thrombosis and pregnancy morbidity as diagnostic manifestations of APS ( 1 ), the new 2023 ACR/EULAR criteria has introduced several well-defined non-thrombotic manifestations into the clinical criteria for APS classification, including microvascular diseases, cardiac valve diseases, and thrombocytopenia ( 2 ). A progression and advancement of the comprehension of aPL-related clinical manifestations is indicated. However, limitations still exist as patients with criteria aPL and comparatively uncommon non-thrombotic manifestations and patients with fulfillment of clinical criteria but seronegative conventional aPL might be inadequately excluded. These conditions are therefore suggested to be referred as “probable APS” or “non-criteria APS” ( 15 ). Our case met the laboratory criteria based on persistence of medium-to-highly positive aCL and LA. The complete and sustained remission of hepatic vasculitis was achieved only when aPL were managed at lower titers with pathogenic effects effectively suppressed. The development of cerebral infarction happened in the setting of highly positive aPL and in the absence of other vascular risk factors. Hepatic vasculitis and cerebral infarction were therefore considered to be associated manifestations. The pathophysiology of cerebral infarcts was undetermined, yet the remarkably elevated D-dimer and FDP suggested a possibility of thrombotic events. Accordingly, our case was assessed as highly probable APS with aPL-related hepatic vasculitis as a non-criteria manifestation, and the development of pathogenic aPL was associated with prior COVID-19 infection.

Infections have been implicated in induction of autoimmunity including aPL production ( 16 ), with the recent COVID-19 pandemic being no exception ( 7 ). A large number of studies have reported high prevalence of aPL (5%–71%), both criteria and non-criteria types, in COVID-19 patients ( 7 , 17 ). Several potential mechanisms have been proposed but require further validation ( 8 ). Molecular mimicry supposes that the S1 and S2 subunits of the SARS-CoV-2 viral S protein might form a phospholipid-like epitope shared with native tissues, triggering aPL production and provoking an immunogenic response ( 18 – 20 ). The neoepitope model posits that oxidative stress induced by SARS-CoV-2 can alter the conformation of β2GPI ( 21 , 22 ) and create a neoepitope for antibody generation ( 23 ).

Despite the observed high prevalence, the pathogenicity of COVID-19-associated aPL remains uncertain and controversial. To explore the potential roles of aPL, numerous studies have analyzed the correlations of aPL and clinical manifestations in COVID-19 patients, yet a consensus could not be reached. COVID-19-associated aPL were demonstrated to be natural or nonpathogenic in most studies ( 24 – 50 ), which was also shown in the largest meta-analysis published in 2021 ( 51 ). Additionally, anti-β2GPI in COVID-19 was reported to rarely (5%) recognize domain I of β2GPI, the molecular region most commonly associated with pathogenicity ( 24 ). On the contrary, associations of aPL with disease severity and thrombosis in COVID-19 patients were also reported ( 52 – 67 ). Notably, the largest cohort study demonstrated a correlation between the presence of aCL or IgA anti-β2GPI and thrombotic events ( 65 ). IgG antibodies purified from COVID-19 patients with high aPL titers were found to trigger neutrophil extracellular trap release and potentiate thrombosis in mice, similarly to IgG isolated from individuals with definite APS ( 59 ). Additionally, infections have been reported as the most common causative factor of catastrophic APS (CAPS), suggesting that infection-induced aPL could exhibit biological activity in a subset of patients ( 6 ). Several theories have been proposed to explain the heterogeneity. The “two hits” theory holds that aPL (first hit) induce a thrombophilic state, but clotting requires additional thrombophilic condition (second hit), often involving an innate immunity activator like inflammation, infection, or surgery ( 3 ). Furthermore, infections are proposed to more likely trigger APS in individuals with genetic propensity, immune defects, or hormonal abnormalities ( 16 ). Therefore, the pathogenicity of aPL exhibits heterogeneity across COVID-19 patients and susceptible individuals with predisposing factors might present aPL-related manifestations in the presence of COVID-19-associated aPL.

Albeit the intense exploration of aPL in COVID-19 patients by multiple studies, most of them neither specify the duration of aPL positivity nor subgroup patients according to antibody levels. All COVID-19 patients with positive aPL were incorporated, and individuals with or without pathogenic aPL were merged together for characterization and analysis, contributing to the debatable pathogenicity of aPL. Systematic analyses based on these studies could not reveal the prevalence and features of patients developing pathogenic aPL after COVID-19. Conversely, new-onset APS in COVID-19 patients have also been reported, wherein persistently high-titer aPL, associated thrombotic and non-thrombotic manifestations, and recovery following treatments based on APS management guidelines substantially indicate the pathogenicity of aPL. Therefore, new-onset APS cases could be a narrow representative of COVID-19 patients with pathogenic aPL. We systematically reviewed the literature of relevant cases up to February 2024 using PubMed and EMBASE to analyze the APS onset after COVID-19 for the first time. The cases reported as APS after COVID-19 infection with sufficient information to meet the updated Sapporo criteria or the new 2023 ACR/EULAR criteria ( 1 , 2 ) or with limited information but compelling evidence to support the diagnosis of APS were included. The former and the latter were annotated as definite APS and highly probable APS respectively. Nine cases ( 68 – 76 ) were identified and evaluated together with our case ( Table 2 ). The patients ranged from 15 to 89 (mean = 41.10) years of age and most of the patients were female, consistent with the epidemiology of APS that is more common in middle-aged women ( 82 ). According to WHO-issued guidelines ( 83 ), COVID-19 severity of these cases encompassed a spectrum from non-severe to critical, indicating that aPL-related manifestations did not merely develop on the basis of cytokine storms in critical patients. The time interval from COVID-19 to the onset of APS varied from 7 to 41 days (mean = 18 days) and a probably more frequent occurrence during the convalescent period was suggested. Definite or probable CAPS was reported in three cases (30%), significantly higher than the approximate 1% incidence of CAPS in all APS patients ( 84 ). Thrombosis and corresponding organ infarctions (80%) were the most common manifestation, followed by thrombocytopenia (30%). There were 12 patients mentioned in four cohort studies who also fulfilled the inclusion criteria but were not included due to the lack of individualized information ( 50 , 64 , 67 , 77 – 81 , 85 ). In addition to newly diagnosed APS, five cases have reported relapses of completely remitted APS following COVID-19 infections ( Table 2 ), reiterating SARS-CoV-2 as a potential trigger for pathogenic effects of aPL and exacerbation of APS in some patients. Here, we report the first case of developing pathogenic aPL in a juvenile after non-severe COVID-19, diagnosed as highly probable APS. Notably, the absence of any medical history in the patient alerts the possibility of developing severe aPL-related symptoms following non-severe COVID-19 infection in previously healthy individuals, which was also indicated in a healthy woman developing obstetric APS (OAPS) after non-severe COVID-19 infection ( 73 ).

Table 2 Demographic and clinical characteristics of APS after COVID-19.

Differences in distribution, clinical presentations, and outcomes exist between pediatric and adult APS ( 86 ). Compared with adult patients, juvenile patients more frequently exhibit non-thrombotic aPL-related manifestations ( 10 ). A study including 121 juveniles fulfilling the updated Sapporo criteria demonstrated a high prevalence of associated non-thrombotic manifestations with neurologic, hematologic, and skin disorders being the most common ( 87 ). Non-thrombotic manifestations sometimes precede later thrombotic events ( 88 ), leaving pediatric patients with isolated non-thrombotic manifestations being inadequately excluded from APS patient population. Accordingly, diagnostic criteria for definite APS are inapplicable in juveniles. Recommendations for management of pediatric APS published by SHARE initiative advocated for the incorporation of non-criteria manifestations into classification criteria for pediatric APS ( 86 ). Therefore, recent studies in pediatric APS have concentrated mainly on pathogenic aPL and associated manifestations rather than definite APS. A study of pediatric APS including definite and probable cases revealed high percentage of hematologic and skin disorders ( 89 ). Moreover, another analysis of children with medium or highly positive aPL suggested that more than half exhibited non-thrombotic aPL-related manifestations alone ( 90 ).

In our case, the histopathology of liver biopsy specimens revealed immune infiltration and fibrinoid necrosis of arteries without granulomatosis, indicating the existence of hepatic vasculitis that has not been reported in association with pathogenic aPL before. The resultant occlusion of arteries gave rise to liver ischemic necrosis in the absence of any notable thrombus or microthrombus. The patient was successfully treated with immunosuppression, further supporting a vasculitic other than thrombotic etiology.

Although debatable, vascular lesions raised by aPL could be inflammatory. DAH, characterized by bleeding into the alveolar space resulting from disruption and injury of pulmonary microcirculation, represents a genuine inflammatory complication of APS and has been included into clinical criteria for APS in the 2023 ACR/EULAR criteria ( 2 , 91 ). Several studies have investigated the primary APS-associated DAH in recent years ( 11 , 92 – 94 ). Surgical or transbronchial biopsies were performed in 20 cases and capillaritis without thrombus or microthrombus was histologically documented in 11 of them (55%), indicating an isolated inflammatory vasculopathy in DAH development. The recommended and efficient treatment of DAH in APS with glucocorticoids and immunomodulatory agents re-emphasizes an inflammatory instead of thrombotic etiopathology of DAH ( 91 ). Additionally, mesenteric vasculitis is considered to be one of aPL-related microvascular manifestations as well ( 95 ). Sporadic cases with authentic associated vasculitic manifestations have also been reported in cerebral ( 96 ), renal ( 97 ), aortic ( 98 ), and cutaneous ( 99 ) vasculature, and no local thrombus or microthrombus was noted in these inflammatory lesions.

Therapy for APS is diverse and individualized based on a broad spectrum of manifestations. Long-term oral anticoagulants like warfarin are recommended for thrombotic APS ( 100 ), and alternative therapies such as extended therapeutic dose of low-molecular-weight heparin can be utilized for patients with recurrent thrombotic events despite warfarin ( 101 ). For aPL carriers with high-risk profiles or OAPS patients, low-dose aspirin is proposed for primary thrombosis prevention, particularly in individuals with additional vascular risk factors ( 100 , 102 ). Glucocorticoids; immunomodulatory agents including MMF, cyclophosphamide, and azathioprine; and B-cell-modulating agents like rituximab and belimumab, are recommended in cases with non-thrombotic manifestations ( 103 , 104 ). Notably, these recommendations, based on adult-derived studies, might be improper for pediatric populations due to differences in physiological conditions, metabolic capacities and duration of medication. Additionally, the low prevalence and heterogeneity of APS in juveniles impede the formation and limit the strength of evidence-based guidelines ( 86 , 105 ), contributing to substantial variations in treatment regimens that are mostly based on physicians’ experience or observational studies.

In our case, aspirin was administered without anticoagulants. The decision was made based on vasculitis-induced hepatic infarction as the major clinical presentation, repair of cerebral lesions with indefinite pathology before systemic treatment, impaired liver synthetic function for coagulation factors, and the absence of other thrombosis risk factors. As concurrent thrombosis risk factors like arterial hypertension, hyperlipidemia, atherosclerosis and smoking are rarely observed in younger subjects, long-term anticoagulation therapy is not indicated in pediatric thrombotic APS patients harboring discontinuous aPL ( 106 – 108 ). Likewise, immunosuppressive therapy in our case reduced aPL titers close to baseline levels and suppressed their pathogenicity, reminiscent of patients with discontinuous aPL. Combined together, anticoagulants were not administered temporarily. However, the patient underwent intensive and regular follow-up to monitor for emergence of any additional thrombosis risk factors, in which scenario, anticoagulants would be introduced as a replacement of aspirin.

aPL-related thrombosis and vasculitis can cause similar clinical presentations including organ infarctions, whereas the treatment decision is different due to the underlying pathologies. The histopathologic results helped us to confirm the inflammatory vasculopathy and guided the treatment to adequately include immunosuppression comprising glucocorticoids and immunomodulatory agents. Therefore, when no thrombus is detected by non-invasive examinations, biopsy for confirmation of the underlying vasculopathy is suggested in APS, if possible and especially when liver is involved.

Our treatments were individualized based on an atypical case. Although the outcome was favorable, the efficacy and safety of aspirin without anticoagulants require further validation during extended follow-up. We merely recommend the addition of immunosuppressants to conventional therapy for managing aPL-related vasculitis.

4 Conclusion

Given the perplexing and contentious nature of aPL produced during infections, the COVID-19 pandemic provides a distinctive opportunity to comprehensively assess this issue. The literature review and analysis evaluate the onset and relapse of APS after COVID-19 infection, suggesting that SARS-CoV-2-triggered aPL may exert pathogenic effects in a subset of COVID-19 patients.

Altogether, we endorse the hypothesis that pathogenic aPL can raise vascular damage manifested as vasculitis other than thrombosis, conveying distinct therapeutic considerations to include immunosuppression therapy. In addition to vasculitis, other forms of vascular lesions including proliferative vascular diseases have also been described in APS ( 109 ), extending the spectrum of vascular changes associated with pathogenic aPL. Such cumulative evidence supports the statement that the nature of APS should be extended to both thrombophilia and vasculopathy.

Data availability statement

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

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Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.

Author contributions

QL: Conceptualization, Formal analysis, Investigation, Writing – original draft. JL: Conceptualization, Formal analysis, Investigation, Writing – original draft. MZ: Writing – review & editing. YG: Writing – review & editing. ZL: Funding acquisition, Writing – review & editing. TL: Funding acquisition, Writing – review & editing. LZ: Resources, Supervision, Writing – review & editing.

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This study was supported by the National High Level Hospital Clinical Research Funding (2022-PUMCH-B-043) and the National Natural Science Foundation of China (82202541).

Conflict of interest

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

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Keywords: antiphospholipid antibodies, COVID-19, pediatrics, vasculitis, non-thrombotic manifestation, vasculopathy

Citation: Li Q, Li J, Zhou M, Ge Y, Liu Z, Li T and Zhang L (2024) Antiphospholipid antibody-related hepatic vasculitis in a juvenile after non-severe COVID-19: a case report and literature review. Front. Immunol. 15:1354349. doi: 10.3389/fimmu.2024.1354349

Received: 12 December 2023; Accepted: 29 March 2024; Published: 19 April 2024.

Reviewed by:

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

*Correspondence: Li Zhang, [email protected]

† These authors have contributed equally to this work and share first authorship

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

• Research Article
• Open access
• Published: 20 January 2024

Dementia care pathways in prisons – a comprehensive scoping review

• Samantha Treacy 1 ,
• Steven Martin 2 ,
• Nelum Samarutilake 3 ,
• Veronica Phillips 4 ,
• Ben R. Underwood 3 , 5 &
• Tine Van Bortel   ORCID: orcid.org/0000-0003-0467-6393 2 , 3  

Health & Justice volume  12 , Article number:  2 ( 2024 ) Cite this article

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The number of older people in prison is growing. As a result, there will also be more prisoners suffering from dementia. The support and management of this population is likely to present multiple challenges to the prison system.

To examine the published literature on the care and supervision of people living in prison with dementia and on transitioning into the community; to identify good practice and recommendations that might inform the development of prison dementia care pathways.

A scoping review methodology was adopted with reporting guided by the PRISMA extension for scoping reviews checklist and explanation.

Sixty-seven papers were included. Most of these were from high income countries, with the majority from the United Kingdom ( n  = 34), followed by the United States ( n  = 15), and Australia ( n  = 12). One further paper was from India.

The literature indicated that there were difficulties across the prison system for people with dementia along the pathway from reception to release and resettlement. These touched upon all aspects of prison life and its environment, including health and social care. A lack of resources and national and regional policies were identified as important barriers, although a number of solutions were also identified in the literature, including the development of locally tailored policies and increased collaboration with the voluntary sector.

To our knowledge, this is the most comprehensive and inclusive review of the literature on dementia care pathways in prison to date. It has identified a number of important areas of concern and opportunities for future research across the prison system, and its operations. This will hopefully lead to the identification or adaptation of interventions to be implemented and evaluated, and facilitate the development of dementia care pathways in prisons.

The number of older people (defined here as those over 50 Footnote 1 ) being held in prison in England and Wales has almost tripled over the last 20 years, and they now represent 17.1% of that population (Ministry of Justice, 2022a ). The growing number of older people has brought with it an increasing number of health and social care problems, reportedly affecting around 85% of older people in prison, with associated costs (Di Lorito, et al., 2018 ; Hayes et al., 2012 , 2013 ; Senior, et al., 2013 ). It has been estimated that 8.1% of those over the age of 50 in prison have mild cognitive impairment or dementia, which is much higher than estimates for this age group in the general population (Dunne et al., 2021 ; Forsyth et al., 2020 ). This pattern of poor health also increased the vulnerability of older people in prison during the pandemic (Kay, 2020 ).

Prison policy and legislation mandates that health and social care be ‘equivalent’ to that provided in the community (Care Act, 2014 ; Department of Health, 1999 ). Despite this, provisions are reportedly inconsistent, and the government has been described as ‘failing’ in its duty of care (Health and Social Care Committee, 2018 ; HM Inspectorate of Prisons & Care Quality Commission, 2018 ). This is likely exacerbated by the suspension and limiting of healthcare services during the pandemic, noted to have had a ‘profound’ impact on people’s health and wellbeing (HM Inspectorate of Prisons, 2021 ). This may be particularly so for people living in prison with dementia (PLiPWD), whereby the difficulties of delivering health and social care are compounded by inappropriate buildings, environments, and prison regimes (rules and regulations). In addition, PLiPWDs may experience an increase in social isolation, including separation from friends and family, all of which may make their time in prison more challenging (Moll, 2013 ; Peacock et al., 2019 ).

There is no current national strategy for older people in prison in England and Wales, including PLiPWD, although the British government recently agreed that there is a need for one (Justice Committee, 2020 ). A ‘Model for Operational Delivery’ for older people has been published by Her Majesty's Prison & Probation Service ( 2018 ) in England and Wales, though this is guidance only and the “properly resourced and coordinated strategy” previously called for has not been produced (Prisons & Probation Ombudsman, 2017 , p7; Brooke and Rybacka, 2020 ; HM Inspectorate of Prisons, 2019 ; Justice Committee, 2020 ). One way of attempting to standardise and improve the quality of treatment and care in the community has been through the use of care pathways (Centre for Policy on Ageing, 2014 ; Schrijvers et al., 2012 ). Care pathways have been defined as “a complex intervention for the mutual decision-making and organisation of care processes for a well-defined group of patients during a well-defined period”, involving an articulation of goals and key aspects of evidence-based care, coordination and sequencing of activities and outcomes evaluation (Vanhaecht, et al., 2007 , p137).

The development of care pathways within the prison system lags behind that of the community, but the National Institute for Health and Care Excellence (NICE) has produced a pathway for prisoner health for England and Wales (National Institute for Health and Care Excellence, 2019 ), and there is a care pathway for older prisoners in Wales (Welsh Government & Ministry of Justice, 2011 ). There has also recently been an overall care pathway developed for people in prison with mild cognitive impairment and dementia, although this has not been implemented as yet, and it does not include any details regarding release and resettlement (Forsyth et al, 2020 ). It has been recommended that care pathways should be developed locally, as they are context-sensitive, should be viewed as processual and flexible, and the needs of the person, their experiences and characteristics need to be taken into account – such as age, gender and race (Centre for Policy on Ageing, 2014 ; Pinder, et al., 2005 ).

Here we review the current literature on people living in prison with dementia. There have been two recent systematic literature reviews conducted on PLiPWD, both of which only included primary research studies that were small in number (Brooke and Rybacka, 2020 ( n  = 10); Peacock et al., 2019 ( n  = 8)), and focused on prevalence, identification (screening and diagnosis), and the need for tailored programming and staff training. Peacock et al., ( 2019 ) identified dementia as a concern and suggested recommendations for improved screening and care practices. Brooke et al. ( 2020 ) noted that, whilst the prevalence of dementia in prison populations was largely unknown, there was a need for national policies and local strategies that support a multi-disciplinary approach to early detection, screening and diagnosis. Neither paper, however, reported on the much more extensive and rich grey literature in this area (Brooke and Rybacka, 2020 ), to help comprehensively identify the systemic and operational problems, barriers and potential solutions that would be useful to consider in developing local dementia care pathways. Therefore, the aim of this paper is to conduct a comprehensive systematic scoping review of the available published literature on the support and management of PLiPWD in prison and upon transitioning into the community, and to identify practice and recommendations that would be useful to consider in the development of a local prison dementia care pathway.

A scoping review methodology using Arksey and O’Malley’s ( 2005 ) five-stage framework was adopted for this review. Reporting was guided by the PRISMA extension for scoping reviews checklist and explanation (Tricco et al., 2018 ). The completed checklist for this review is available in Additional file 1 : Appendix 1.

Identification of relevant reports

The search strategy was formulated by the research team, and included an electronic database search and subsequent hand search. The electronic search involved searching twelve electronic databases: Applied Social Sciences Index and Abstract, Criminal Justice Abstracts, Embase, Medline (OVID), National Criminal Justice Reference Service, Open Grey, Psycinfo, Pubmed, Scopus, Social Services Abstracts, Sociological Abstracts, and Web of Science. The search combined condition-related terms (dementia OR Alzheimer*) AND context-related ones (prison OR jail OR gaol OR penitentia* OR penal OR correctional* OR incarcerat*), with no date or language restrictions, and covered the full range of publications up until April 2022. Additional file 2 : Appendix 2 has an example of the search strategy used.

Electronic searches were supplemented by comprehensive hand searching and reference mining. Searches were also undertaken using: search engines; websites related to prisons and/or dementia (for example, Prison Reform Trust); a database from a previous related literature review (Lee et al, 2019 ); recommendations from academic networking sites; contacting prominent authors in the field directly; government-related websites (for example Public Health England, now called Health Security Agency); recent inspection reports for all prisons in England and Wales from Her Majesty’s Inspectorate of Prisons and the Independent Monitoring Board.

Inclusion and exclusion criteria

Papers were considered suitable for inclusion in this review if they met the following criteria:

Setting: Papers should primarily be set in, or pertain to, prisons. Documents solely referring to community services, hospitals or medical facilities that are not part of the prison system were excluded.

People: Papers involving PLiPWD. Research focused only on older people in prison more generally was excluded, as was research which described the disorienting effects of imprisonment more generally, but which was not related to dementia.

Intervention: Some consideration of the treatment, care, support or management of PLiPWD; this can be health or social-care associated, as well as related to the prison overall, and to any individuals, groups or agencies who visit or work with individuals during their time in prison (including family, friends, charities, probation services). Papers which mostly describe prevalence studies, sentencing practices or profiles were excluded.

Study design: All designs were considered for inclusion. Editorials, book reviews, online blogs, press releases, announcements, summaries, newspaper and magazine articles, abstracts and letters were excluded.

The titles, abstracts and full-text of the papers identified by the searches were screened for inclusion in the review. The screening was undertaken by two independent researchers (ST and NS) for inter-rater reliability purposes (Rutter et al., 2010 ). Any differences of opinion on inclusion were resolved between the researchers (ST, NS and SM), and with the Principle Investigator (TVB).

Charting the data

An extraction template was developed for the review, guided by the PICO formula (Richardson et al., 1995 ) and informed by pathway stages and key areas highlighted in the older prisoner pathways toolkit for England and Wales (Department of Health, 2007 ), and the older prisoner pathway formulated for Wales (Welsh Government & Ministry of Justice, 2011 ). Using this extraction template, all of the data was extracted from the included papers by one member of the research team (ST), with a second researcher extracting data from a third of the papers as a check for consistency (SM). Any unresolved issues were related to the Principle Investigator (TVB) for resolution.

Collating, summarising and reporting results

The review was deliberately inclusive of a wide variety of types of papers, which meant that taking a meta-analytic approach to the data was not feasible. Therefore, a narrative approach to summarising and synthesising the findings and recommendations of the included papers was adopted (Popay et al, 2006 ).

Sixty-seven papers were included in this scoping review. The screening process phases conducted by the research team are shown in Fig.  1 .

PRISMA flow diagram

A brief overview of the key features of each of the papers is presented in Table 1 . All but one of the included papers were from high income countries, with the majority from the United Kingdom ( n  = 34), and then the United States ( n  = 15), Australia ( n  = 12), Canada ( n  = 4), Italy ( n  = 1) and India ( n  = 1). The papers were split into types, with twenty-two guidance and inspection documents, and twenty-seven discussion and intervention description papers. Of the eighteen research and review articles with a defined methodology included there were four literature reviews (one was systematic), nine qualitative studies, four mixed-methods studies (one which followed participants up), and one survey-based study.

Areas to consider in the support and management of PLiPWD during their time in prison and upon their release

The pathway through the prison is shown in Fig.  2 , and typically involves: (i) reception into prison; (ii) assessments, and allocation of the person within prison; (iii) time held in prison; (iv) transfers between prisons, and between prisons and other services such as time spent in hospital; and (v) release and preparations for resettlement in the community. There were also a number of (vi) cross-cutting themes which could potentially impact people with dementia living in prison at each stage across the prison pathway.

Dementia prison pathway considerations

(i) Reception

Upon entry into prison, prisoners are subject to an initial reception screening to identify and support immediate health and social care problems, and those in need of further assessment. An induction to prison rules and regulations also typically occurs at this step.

All papers reported that reception screening with appropriate screening tools was important in identifying cognitive difficulties and in establishing a baseline, but implementation seemed to vary (Peacock et al., 2019 ). One study in England and Wales found only 30% of prisons contacted routinely did this (Forsyth et al., 2020 ). Supporting policy and a service/person to refer to directly for further assessment were also highlighted as useful (Brooke & Jackson, 2019 ; Brooke et al., 2018 ; Gaston & Axford, 2018 ; Inspector of Custodial Services, 2015 ; Patterson et al., 2016 ). Proposed cut-offs for this screening were either 50 years of age ( n  = 7), under 55 years ( n  = 1), or 55 years of age ( n  = 7). One paper reported that only a third of prisoners who were offered this screening accepted it, although the reasons for this were not stated (Patel & Bonner, 2016 ). Another paper suggested that a screening programme could have unintended adverse consequences, that could damage already fragile relationships between staff and people living in prison (Moore & Burtonwood, 2019 ). Whilst many screening tools were mentioned, there are currently no tools validated for use in prisons, and many of those used in the community may be inappropriate (Baldwin & Leete, 2012 ; Brooke et al., 2018 ; du Toit et al., 2019 ; Feczko, 2014 ; Forsyth et al., 2020 ; Moore & Burtonwood, 2019 ; National Institute for Health and Care Excellence, 2017 ; Turner, 2018 ; Williams et al., 2012 ). One validation study found that the Six-item Cognitive Impairment Test (6CIT) was not suitably sensitive for use (Forsyth et al., 2020 ). Other difficulties included the limited amount of time and resources available to screen at reception (Christodoulou, 2012 ; Patterson et al., 2016 ; Peacock et al., 2019 ), and that staff lacked ‘familiarity’ with screening tools (Peacock et al., 2019 ).

Only two papers mentioned the induction process (Her Majesty's Prison & Probation Service, 2018 ; Welsh Government and Ministry of Justice, 2011 ) as important. A need for clearly explained information in a dementia-appropriate format (written and verbal) particularly regarding healthcare, and a recommendation that PLiPWD should be regularly reminded of rules and regulations, were suggested.

(ii) Assessment

Following the screening process, the current recommendation is that an initial healthcare assessment takes place in the first seven days after entering prison. During this initial assessment period, although not necessarily within this timeframe, care plans and allocation decisions may also be made regarding where the prisoner is placed within the prison.

An initial older-person-specific health and/or social care assessment or standard process for assessment has been recommended by ten papers, six of which were from government or related bodies. It was also suggested by some papers, that a cognitive assessment should take place at either 50 years ( n  = 6) or 55 years ( n  = 2), which should be repeated every three months ( n  = 3), six months ( n  = 5) or annually ( n  = 12), with the latter including recommendations from NICE guidelines (National Institute for Health and Care Excellence, 2017 ). One study set in England and Wales found that most prisons (60%) that screened older people, did so between 7–12 months (Forsyth et al., 2020 ). Brief and affordable tools were considered more useful (Garavito, 2020 ; Turner, 2018 ), although the Montreal Cognitive Assessment (MOCA) was recommended in the care pathway developed by Forsyth et al. ( 2020 ).

Typically, assessments were conducted by healthcare staff, GPs or a psychologist ( n  = 6), a specialist in-house assessment unit ( n  = 2), or a specific dementia admissions assessment unit ( n  = 4). For further assessment, some prisons had internal teams to refer to ( n  = 5). Forsyth et al. ( 2020 ) recommend referral to external Memory Assessment Services for assessment. A case finding tool was being piloted in one prison (Sindano & Swapp, 2019 ). Assessments included can be found in Table 2 .

Assessments also explored risk and safeguarding (National Institute for Health and Care Excellence, 2017 ; Patterson et al., 2016 ; Welsh Government and Ministry of Justice, 2011 ), environmental impact (National Institute for Health and Care Excellence, 2017 ), capacity (Prison & Probation Ombudsman, 2016 ), work, education, and drug and alcohol use (Welsh Government and Ministry of Justice, 2011 ) and a person’s strengths (Hamada, 2015 ; National Institute for Health and Care Excellence, 2017 ). Prison staff contributed to some assessments of activities of daily living (ADLs) or prison-modified ADLs (Brooke et al., 2018 ; Brown, 2016 ; Dillon et al., 2019 ; Department of Health, 2007 ; Feczko, 2014 ; Forsyth et al., 2020 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Patterson et al., 2016 ; Turner, 2018 ; Welsh Government and Ministry of Justice, 2011 ; Williams et al., 2012 ). Challenges to Assessment can be found in Table 3 .

Twelve papers described or recommended care planning post-assessment, in collaboration with PLiPWD and primary care, or a multi-disciplinary team (MDT) of health, social care and prison staff with external specialists healthcare proxies charities or family (Brown, 2016 ; Dillon et al., 2019 ; du Toit & Ng, 2022 ; Hamada, 2015 ; Her Majesty's Inspectorate of Prisons, 2014 ; Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ; National Institute for Health and Care Excellence, 2017 ; Patterson et al., 2016 ; Prisons and Probation Ombudsman, 2016 ; Welsh Government and Ministry of Justice, 2011 ). However, it was suggested that prison staff be removed from the decision-making process as the dementia progresses, and be part of the ‘duty of care’ of healthcare staff and services (du Toit & Ng, 2022 ). It was recommended too that care plans be disseminated to prison wing staff (Forsyth et al., 2020 ) and peer supporters (Goulding, 2013 ), and that consent be sought for this (Goulding, 2013 ; Her Majesty's Inspectorate of Prisons, 2014 ) An ombudsman report in England and Wales noted that care plans for PLiPWD who had died in prison were inadequate (Peacock et al., 2018 ), and of the varying degrees of care planning found by Forsyth et al ( 2020 ), it was described typically as “rudimentary” (p26). Care plans are described further in Table 4 .

Many papers reported that prisons did or should make decisions about where people should be accommodated within the prison after health assessments (Brown, 2016 ; Feczko, 2014 ; Forsyth et al., 2020 ; Hodel & Sanchez, 2013 ; Inspector of Custodial Services, 2015 ; Mistry & Muhammad, 2015 ; Turner, 2018 ; Welsh Government and Ministry of Justice, 2011 ; Williams et al., 2012 ), taking age and health into account. However, despite recommendations that PLiPWD should be placed on the ground floor on low bunks for instance (Baldwin & Leete, 2012 ; Department of Health, 2007 ; Welsh Government and Ministry of Justice, 2011 ), there were reports that this was not happening (Inspector of Custodial Services, 2015 ). There were also recommendations for allocations to be made across a region to ensure people are appropriately placed in the prison system (Baldwin & Leete, 2012 ; Booth, 2016 ; Gaston & Axford, 2018 ; Welsh Government and Ministry of Justice, 2011 ). Concerns were expressed about the lack of lower category places for PLiPWD (Department of Health, 2007 ), and the lack of guidance regarding placement of people with high support needs (Sindano & Swapp, 2019 ) in England and Wales.

(iii) Within-prison issues

A number of papers reported on a need for policies or frameworks to support staff to identify, assess and support people who may be living with dementia (Brooke et al., 2018 ; Brooke & Jackson, 2019 ; Department of Health, 2007 ; Feczko, 2014 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Patterson et al., 2016 ; Turner, 2018 ; Welsh Government and Ministry of Justice, 2011 ), without which staff have faced difficulties in providing quality care and support (Feczko, 2014 ; Prisons and Probation Ombudsman, 2016 ). Whilst there were some examples of guidance for dementia (Hamada, 2015 ; Patterson et al., 2016 ; Treacy et al., 2019 ; Turner, 2018 ), it was suggested that all policies should be reviewed and amended to ensure that they are appropriate for older people and people living with dementia (Department of Health,  2007 ; Lee et al., 2019 ; Treacy et al., 2019 ). Specific policy areas are described in Table 5 .

Issues around staff training on dementia were discussed in the majority of papers ( n  = 54) Many of these reported that prison staff either lacked training on dementia, or that training was limited ( n  = 16), with one study in England and Wales reporting that only a quarter of prison staff had received such training (Forsyth et al., 2020 ). Perhaps consequently, a number of papers identified that prison staff required some dementia training ( n  = 19). Staff working on a specialist dementia unit reportedly had a comprehensive 40-h training (Brown, 2014 , 2016 ; Gaston & Axford, 2018 ; Hodel & Sanchez, 2013 ; Moll, 2013 ), and it was suggested that more comprehensive training be facilitated for officers, particularly those working with PLiPWD ( n  = 18) and offender managers ( n  = 2). A need for all staff working with PLiPWD to be supervised was also suggested (Gaston & Axford, 2018 ; Maschi et al., 2012 ). Despite a lack of consensus on content and duration (du Toit et al, 2019 ), typically, the staff training undertaken and recommended was in four areas (Table 6 ). It was also recommended that training for healthcare could be more comprehensive and focused on screening, identification, assessment, diagnoses, supervision and intervention training (Baldwin & Leete, 2012 ; Brooke & Jackson, 2019 ; Brown, 2014 ; Gaston & Axford, 2018 ; Her Majesty's Inspectorate of Prisons, 2014 ; Moll, 2013 ; Moore & Burtonwood, 2019 ; National Institute for Health and Care Excellence, 2017 ; Peacock et al, 2019 ; Treacy et al, 2019 ; Turner, 2018 ; Williams, 2014 ). It is of note that only 21% of healthcare staff in one study in England and Wales reported attending training to identify dementia (Forsyth et al., 2020 ), similar to the figures regarding prison staff in the same study.

Much of the training described in the included papers had been formulated and delivered by dementia- or older people-specific voluntary organisations (Alzheimer’s Society, 2018 ; Brooke et al.  2018 ; Brown, 2016 ; Gaston & Axford, 2018 ; HMP Hull, 2015 ; Her Majesty's Prison & Probation Service, 2018 ; Hodel & Sanchez, 2013 ; Moll, 2013 ; Peacock et al., 2018 ; Prisons and Probation Ombudsman, 2016 ; Sindano & Swapp, 2019 ; Tilsed, 2019 ; Treacy et al., 2019 ). Although it has also been recommended to involve health and social care (Goulding, 2013 ; Her Majesty's Prison & Probation Service, 2018 ; Ministry of Justice, 2013 ; Treacy et al., 2019 ; Turner, 2018 ), and officers and peer supporters (Brooke & Jackson, 2019 ; Masters et al., 2016 ; National Institute for Health and Care Excellence, 2017 ; Treacy et al., 2019 ) in developing the training. In one study, prison staff were also trained to deliver dementia information sessions to their peers (Treacy et al., 2019 ). A suggestion of video-training packages was also made (du Toit et al., 2019 ). Dementia training typically lacked robust evaluation (Brooke et al., 2018 ), although those available generally reported benefits in their understanding of dementia, relationships, and diagnoses (Goulding, 2013 ; HMP Littlehey, 2016 ; Masters et al., 2016 ; Sindano & Swapp, 2019 ; Treacy et al., 2019 ). It was also reported that some prison staff were resistant to working with PLiPWD (Moll, 2013 ), and that resource limitations resulted in training cuts (HMP Hull, 2015 ; Treacy et al., 2019 ).

Offering healthcare across the spectrum for PLiPWDs, from acute to chronic care, with a focus on preventative and long-term care as well as palliative care was recommended by some papers (Brown, 2014 ; du Toit & Ng, 2022 ; Gaston, 2018 ; Maschi et al., 2012 ; Mistry & Muhammad, 2015 ; Peacock et al, 2018 ; Welsh Government and Ministry of Justice, 2011 ; Williams et al., 2012 ). The development of care pathways to guide this were also recommended or formulated (du Toit et al., 2019 ; Forsyth et al., 2020 ; Peacock et al., 2019 ), although the majority (69%) of prisons in one study in England and Wales did not have one (Forsyth et al., 2020 ). Clear and formal links with local hospitals, memory clinics, forensic and community teams for planning, training, advice, support and in-reach were also present or recommended by sixteen research and guidance papers. The amount of healthcare cover in prisons in England and Wales reportedly varied with the function of the prison with largely only local prisons having 24-h healthcare staff (Treacy et al., 2019 ), and most other forms of prison having office-type hours’ healthcare cover – including sex offender prisons where the majority of older prisoners are held (Brown, 2016 ; Correctional Investigator Canada, 2019 ; Goulding, 2013 ; Inspector of Custodial Services, 2015 ; Treacy et al., 2019 ). While specialist services or units for PLiPWD exist in a number of jurisdictions (Baldwin & Leete, 2012 ; Brown, 2016 ; Cipriani et al., 2017 ; Gaston & Axford, 2018 ; Goulding, 2013 ; Hodel & Sanchez, 2013 ; Inspector of Custodial Services, 2015 ; Maschi et al., 2012 ; Mistry & Muhammad, 2015 ; Treacy et al, 2019 ), more are reportedly needed (Brooke et al., 2018 ; du Toit et al., 2019 ; Forsyth et al., 2020 ; Welsh Government and Ministry of Justice, 2011 ).

Most healthcare teams were reportedly MDT, or this was recommended, alongside joint health and social care working ( n  = 16). A number of healthcare staff acted as the lead for older people in prisons (Department of Health, 2007 ; Her Majesty's Inspectorate of Prisons, 2014 ; Her Majesty's Inspectorate of Prisons, 2016 ; Moll, 2013 ; Welsh Government and Ministry of Justice, 2011 ), with a recommendation that a dementia-trained nurse should lead any dementia care pathways (Forsyth et al., 2020 ) and indeed it was suggested that healthcare staff in general have training and experience in working with older people (Her Majesty's Inspectorate of Prisons, 2014 ; Her Majesty's Inspectorate of Prisons, 2017b ; Moll, 2013 ; Patterson et al., 2016 ; Public Health England, 2017b ; Treacy et al., 2019 ; Turner, 2018 ; Welsh Government and Ministry of Justice, 2011 ). Whilst one of the recommended roles for healthcare was the prescription and monitoring of medication (Feczko, 2014 ; Her Majesty's Inspectorate of Prisons, 2017b ; Moll, 2013 ), much of the focus was on early identification and diagnosis, and keeping a dementia register (Department of Health, 2007 ; Moll, 2013 ; Patterson et al., 2016 ; Welsh Government and Ministry of Justice, 2011 ), and the use of non-pharmacological approaches. These broadly included: psychological interventions (Goulding, 2013 ; Hamada, 2015 ; Moll, 2013 ; Wilson & Barboza, 2010 ); assistance with ADLs and social care (Feczko, 2014 ; Hamada, 2015 ; Hodel & Sanchez, 2013 ; Maschi, et al., 2012 ; Murray, 2004 ; Prisons and Probation Ombudsman, 2016 ); development and delivery of specialist dementia prison programmes (Brown, 2014 , 2016 ; Hodel & Sanchez, 2013 ; Mistry & Muhammad, 2015 ; Moll, 2013 ; Peacock et al., 2018 ; Wilson & Barboza, 2010 ); reablement and rehabilitation (Welsh Government and Ministry of Justice, 2011 ); relaxation (Wilson & Barboza, 2010 ); safeguarding (Hodel & Sanchez, 2013 ); and cognitive stimulation groups (Moll, 2013 ; Williams, 2014 ). Other possible roles included: training or supporting staff and peer supporters, as reported in fourteen papers, as well as advocacy (Feczko, 2014 ; Peacock et al., 2018 ; Welsh Government and Ministry of Justice, 2011 ), allocation, assessment for offending behaviour groups, risk assessments and disciplinary hearings (Booth, 2016 ; Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Murray, 2004 ; Prisons and Probation Ombudsman, 2016 ). Challenges to Healthcare are noted in Table 7 .

Palliative care

A care pathway for dying people that meets community standards was recommended (Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Welsh Government and Ministry of Justice, 2011 ), as was ensuring that people could choose a preferred place to die (Her Majesty's Prison & Probation Service, 2018 ). Some prisoners were moved to community hospices or hospitals (Brooke & Jackson, 2019 ; Inspector of Custodial Services, 2015 ), or it was felt that they should be (Her Majesty's Prison & Probation Service, 2018 ). Although it was noted that some prisons lack relationships with community hospices or palliative care services and need to foster them (Brooke & Jackson, 2019 ; Brown, 2016 ; Correctional Investigator Canada, 2019 ; Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ).

A number of prisons also reportedly had hospices, particularly in the United States (Brooke et al., 2018 ; Brown, 2016 ; Feczko, 2014 ; Goulding, 2013 ; Williams et al., 2012 ), although these have not been comprehensively evaluated (Williams et al., 2012 ). It was recommended that these be staffed by MDTs (Her Majesty's Prison & Probation Service, 2018 ), including chaplains and nutritionists (Her Majesty's Prison & Probation Service, 2018 ; Goulding, 2013 ), and many included prisoner peer supporters (Brooke et al., 2018 ; Goulding, 2013 ). The use of independent contractors was also suggested as staff-prisoner relationships were considered problematic in some prisons (Williams et al., 2012 ). Regarding family, many hospices were described as allowing more visits (Brooke & Jackson, 2019 ; Goulding, 2013 ; Her Majesty's Prison & Probation Service, 2018 ), including one prison with family accommodation (Her Majesty's Prison & Probation Service, 2018 ). Whilst re-engaging with family was reportedly encouraged (Brown, 2016 ), a lack of support was noted (Correctional Investigator Canada, 2019 ). Suggested improvements include a family liaison officer, providing a list of counselling options, and hosting memorial services (Her Majesty's Prison & Probation Service, 2018 ).

Social care

A social care strategy for older prisoners and a social care lead for all prisons in England and Wales has been recommended (Department of Health, 2007 ; Prisons and Probation Ombudsman, 2016 ). It was reported that MDTs working with PLiPWD should and increasingly do include social workers including specialist units and hospices (Baldwin & Leete, 2012 ; Brooke et al., 2018 ; Brown, 2016 ; Cipriani et al., 2017 ; Goulding, 2013 ; HMP Littlehey, 2016 ; Her Majesty's Prison & Probation Service, 2018 ; Maschi et al., 2012 ; Prisons and Probation Ombudsman, 2016 ; Sindano & Swapp, 2019 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ). Social care roles can be found in Table 8 .

The work may be direct or may be through co-ordinating external agencies or peer supporters (Brooke & Jackson, 2019 ; Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Prisons and Probation Ombudsman, 2016 ; Tilsed, 2019 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ). Clarity in these roles was considered paramount, particularly as uncertainty reportedly continues to exist over who is responsible for meeting prisoners’ social care needs in some prisons in England and Wales despite the passing of the Care Act, 2014 (Dementia Action Alliance, 2017 ; Tilsed, 2019 ; Welsh Government and Ministry of Justice, 2011 ). There was also some ambiguity around the threshold PLiPWD were expected to meet in order to access social care (Forsyth et al., 2020 ). In some instances, personal care was delivered informally by untrained and unsupported prison staff and peer supporters in lieu of suitably trained social care workers (Treacy et al., 2019 ), with issues raised about the unavailability of social care through the night (Forsyth et al., 2020 ). Where social care staff were involved in coordinating personal care for prisoners, it was reported as positive for prisoners and prison staff (Her Majesty's Inspectorate of Prisons, 2016 ; Treacy et al., 2019 ), particularly, in one prison, where social care staff were prison-based (Forsyth et al., 2020 ).

Peer supporters

Prisoner peer supporters were operating in a number of prisons, as reported in 22 papers, and their employment was recommended by a further fourteen. Typically, these were people who had ‘good’ disciplinary and mental health records, and certainly in the US, were longer-serving prisoners. A number of papers indicated the need for peer supporters to receive training in dementia, including awareness and support (Brooke et al., 2018 ; Brooke & Jackson, 2019 ; Brown, 2016 ; Correctional Investigator Canada, 2019 ; Department of Health, 2007 ; Dillon et al., 2019 ; du Toit & Ng, 2022 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Goulding, 2013 ; HMP Hull, 2015 ; HMP Littlehey, 2016 ; Her Majesty's Prison & Probation Service, 2018 ; Inspector of Custodial Services, 2015 ; Maschi et al., 2012 ; Mistry & Muhammad, 2015 ; Sindano & Swapp, 2019 ; Tilsed, 2019 ; Treacy et al., 2019 ). Comprehensive 36–40 h training on dementia was delivered for those working on specialist units, including one leading to a qualification (Brooke & Jackson, 2019 ; Brown, 2016 ; Gaston & Axford, 2018 ; Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ). Much of the training was developed and delivered by charities, particularly dementia-related ones, as reported in eleven papers. Ongoing support and supervision was offered or recommended by some prisons, provided largely by health or social care staff or charities (Brooke & Jackson, 2019 ; Brown, 2016 ; Correctional Investigator Canada, 2019 ; Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Gaston & Axford, 2018 ; Maschi et al., 2012 ; Prisons and Probation Ombudsman, 2016 ; Sindano & Swapp, 2019 ; Treacy et al., 2019 ), with informal peer-to-peer support also described (Brown, 2016 ; Gaston & Axford, 2018 ; Treacy et al., 2019 ). The support and supervision received was found to be valuable (Brooke & Jackson, 2019 ; Brown, 2016 ; Treacy et al., 2019 ). Peer-supporter roles are listed in Table 9 .

A number of benefits to: (a) the peer supporters, (b) the prisoners they supported and, (c) the prison, were described, although formal evaluations were lacking (Brown, 2016 ; Christodoulou, 2012 ; Department of Health, 2007 ; du Toit et al., 2019 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Goulding, 2013 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ). This included: payment, development of skills which could be used on release, positive impact on progression through the system, and on self-confidence and compassion, and the creation of a more humane environment. However, frustration and distress amongst peer supporters largely when untrained and unsupported was also reported (Brooke & Jackson, 2019 ; Brown, 2016 ; Correctional Investigator Canada, 2019 ; Inspector of Custodial Services, 2015 ; Prisons and Probation Ombudsman, 2016 ; Treacy et al., 2019 ), and concerns raised in relation to an over-reliance on peers to do work that it is the statutory duty of health and social care to provide (Prisons and Probation Ombudsman, 2016 ; Treacy et al., 2019 ). This was a particular problem in light of personal care being prohibited for peer supporters in England and Wales (Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ). It is also of note that the role of peer supporter may also attract the opprobrium of other prisoners, with reports that they have been seen as ‘snitches’ or ‘dogs’ in some areas (Brown, 2016 ; Goulding, 2013 ). In addition, in some prisons, the peer supporter role was not advocated due to: fear of litigation; fear of replacing staff with peers; belief that people should be acquiring more transferable skills, since many would be unable to undertake care work in the community due to their offence history (Brown, 2016 ; Goulding, 2013 ).

Accommodation

There were mixed views regarding accommodation for PLiPWD. A continuum of prison accommodation was suggested from independent to 24-h care (including assisted living) (Forsyth et al., 2020 ; Gaston & Axford, 2018 ; Williams et al., 2012 ). A number of papers ( n  = 18) recommended that there should be some form of alternative, more appropriate accommodation developed, potentially regional, including secure facilities possibly with a palliative orientation (Hodel & Sanchez, 2013 ; Mistry & Muhammad, 2015 ; Sfera et al., 2014 ). However, there were concerns about the availability, costs and staffing of specialist units, and distances that family would have to travel to visit despite potential benefits (du Toit et al., 2019 ; Moore & Burtonwood, 2019 ). It was also suggested that PLiPWD should be released to live in the community instead (Correctional Investigator Canada, 2019 ).

Within prisons, there was a debate evident within the papers about whether PLiPWD should be accommodated in separate units or integrated within the general prison population, which had generated little clear evidence and mixed views (Brooke & Jackson, 2019 ; Dillon et al., 2019 ; Her Majesty's Prison & Probation Service, 2018 ; Treacy et al., 2019 ). Authors have suggested that specialist or separate wings focused on older people or those with dementia were safer, met peoples’ needs better, and offered better care, support and programmes than integrated units (Brown, 2014 ; Dillon et al., 2019 ; du Toit & Ng, 2022 ; du Toit et al., 2019 ; Goulding, 2013 ; Maschi et al., 2012 ; Murray, 2004 ; Treacy et al., 2019 ; Williams et al., 2012 ), as long as they were ‘opt-in’ for prisoners and staff (Correctional Investigator Canada, 2019 ; Moll, 2013 ; Treacy et al., 2019 ; Williams et al., 2012 ), and opportunities to get off the wing to socialise with others are provided (Treacy et al., 2019 ). The types of ‘specialist’ accommodation that PLiPWD were living in are reported in Table 10 . It is of note that papers reported a highly limited number of beds available in specialist units (Inspector of Custodial Services, 2015 ; Patterson et al., 2016 ; Turner, 2018 ), and that a number of older prisoner-specific prisons were being closed due to costs (Turner, 2018 ).

Four papers described the benefits of older people and those PLiPWD residing within the general prison population (Dillon et al., 2019 ; Her Majesty's Prison & Probation Service, 2018 ; Treacy et al., 2019 ; Williams et al., 2012 ). Those living with dementia reported a benefit from socialising with, and being cared for by, younger people (Dillon et al., 2019 ; Her Majesty's Prison & Probation Service, 2018 ; Williams et al., 2012 ). The presence of older people also reportedly calmed younger prisoners (Dillon et al., 2019 ; Her Majesty's Prison & Probation Service, 2018 ; Williams et al., 2012 ). Importantly, removing people from their prison social networks may have a detrimental effect (Williams et al., 2012 ), and living on specialist units can be stigmatising (Treacy et al., 2019 ).

Regime and activities

The maintenance of prisons regimes is the primary focus of prison officers (Brooke & Jackson, 2019 ). However, there was a reported need ( n  = 19) for PLiPWD to have equal access to activities and services including work, education, gym, library and day centres where they exist, as well as a structured and varied regime on the wing on which they were accommodated, and support to access these. This support could include providing adequate seating (Welsh Government and Ministry of Justice, 2011 ), or giving prisoners more time to accomplish activities, and to assist if needed (Brooke & Jackson, 2019 ; Goulding, 2013 ; Her Majesty's Prison & Probation Service, 2018 ; Hodel & Sanchez, 2013 ). Other recommendations included an overall relaxation of regimes (Gaston & Axford, 2018 ; Treacy et al., 2019 ), an ‘open door’ policy (Brown, 2016 ; Cipriani et al., 2017 ; Goulding, 2013 ; Her Majesty's Inspectorate of Prisons, 2014 ; Her Majesty's Inspectorate of Prisons, 2017b ; Her Majesty's Prison & Probation Service, 2018 ; Treacy et al., 2019 ), more visible staff (The King's Fund, 2013 ), and creating a more communal social environment (Christodoulou, 2012 ). On-wing social activities are described in Table 11 .

Having on-wing work available or alternative means for prisoners who are unable to work to make money was also reportedly important (Christodoulou, 2012 ; Department of Health, 2007 ; Gaston, 2018 ; Gaston and Axford, 2018 ; Her Majesty's Inspectorate of Prisons, 2014 , 2016 , 2017b ; Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ; Murray, 2004 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ). It was suggested that people with dementia should have the chance to work if wanted, and adaptations could be made to work programmes or working days made shorter to facilitate this. Some prisons had specific roles which involved lighter, simple, repetitive tasks such as gardening (Baldwin & Leete, 2012 ; Brooke & Jackson, 2019 ; Inspector of Custodial Services, 2015 ; Moll, 2013 ; Treacy et al., 2019 ). Day centres existed in some prisons, or were thought to be feasible (Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ), and it was suggested that attendance at these could constitute meaningful paid activity (Her Majesty's Prison & Probation Service, 2018 ). The centres were largely developed and facilitated by charities, and ran a wide variety of social, therapeutic, recreational, arts and advice-centred activities (Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ).

Equal access to educational activities, including rehabilitation and offending behaviour programmes, was highlighted as important, particularly where attendance is needed to facilitate people’s progression through the system (Booth, 2016 ; Brooke & Jackson, 2019 ; Dillon et al., 2019 ; Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ). Some prisons provided, or felt there was a need for, particular educational activities for PLiPWD and adaptations may be, or have been, made to learning materials and equipment, content and pace (Brooke & Jackson, 2019 ; Department of Health, 2007 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Her Majesty's Prison & Probation Service, 2018 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ). Dedicated library sessions have been designated in some prisons, and some libraries can and do stock specialist resources including books, audiobooks, reminiscence packs and archives of local photos, music and DVDs (Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ;Treacy et al., 2019 ; Williams, 2014 ). Educational materials could and have been available between sessions to aid memory with distance learning also possible (Brooke & Jackson, 2019 ; Her Majesty's Prison & Probation Service, 2018 ). Suggestions for alternatives for PLiPWD focused on activity and stimulation (du Toit & Ng, 2022 ; Gaston, 2018 ; Her Majesty's Prison & Probation Service, 2018 ), preparing for retirement classes (Department of Health, 2007 ), health promotion (Brooke et al., 2018 ; Christodoulou, 2012 ; Gaston & Axford, 2018 ; Her Majesty's Prison & Probation Service, 2018 ; Maschiet al., 2012 ; Murray, 2004 ; Welsh Government and Ministry of Justice, 2011 ), the arts (Brooke & Jackson, 2019 ) and IT classes (Her Majesty's Prison & Probation Service, 2018 ). Prisoner forums or representative could also be consulted regarding regimes and activities (Moll, 2013 ; Her Majesty's Prison & Probation Service, 2018 ; Welsh Government and Ministry of Justice, 2011 ). Challenges to regimen and activities are described in Table 12 .

Environment

A large number ( n  = 42) of the included papers discussed changes that prisons had made, or should make, to the built environment in order to be more suitable for PLiPWD – in one study in England and Wales, around half of prisons surveyed had made such environmental modifications (Forsyth et al., 2020 ). These focused on: (i) prisoners’ cells, (ii) bathrooms, (iii) dining hall, (iv) outside space and recreation areas, and (v) overall general prison environment (Table 13 ).

Problematically, the age and dementia-inappropriateness of buildings are considered a challenge (Baldwin & Leete, 2012 ; Brown, 2016 ; Dementia Action Alliance, 2017 ; Forsyth et al., 2020 ; Goulding, 2013 ; Inspector of Custodial Services, 2015 ; Mistry & Muhammad, 2015 ; Prisons and Probation Ombudsman, 2016 ; Treacy et al., 2019 ). Difficulties in navigating prisons where everywhere looks the same (Dementia Action Alliance, 2017 ; Murray, 2004 ; Treacy et al., 2019 ), and the lack of budget (HMP Littlehey, 2016 ; HMP Littlehey, 2016 ; Inspector of Custodial Services, 2015 ; Treacy et al., 2019 ) were also reported issues. It was suggested that the use of dementia-friendly environmental checklists could be useful, potentially with input from occupational therapists, health and social care, and dementia charities and in-house education, work and estates departments (Brown, 2014 ; Christodoulou, 2012 ; Dillon et al., 2019 ; Goulding, 2013 ; HMP Littlehey, 2016 ; Her Majesty's Prison & Probation Service, 2018 ; Hodel & Sanchez, 2013 ; Peacock et al., 2018 ; Sindano & Swapp, 2019 ; Treacy et al., 2019 ). Hope was expressed that newly built prisons would be more dementia-friendly (Dementia Action Alliance, 2017 ; Her Majesty's Prison & Probation Service, 2018 ; Williams et al., 2012 ).

Formal policies and procedures should be in place to help maintain links between family and prisoners, and to foster an understanding of the central importance of families particularly for PLiPWD (Her Majesty's Inspectorate of Prisons, 2016 ; Treacy et al., 2019 ). Some papers described how prisons could support contact by: giving help and additional time to make telephone calls and arranging visits in quieter spaces (Her Majesty's Prison & Probation Service, 2018 ; Prisons and Probation Ombudsman, 2016 ; Treacy et al., 2019 ); increasing the number of visits (Jennings, 2009 ); and allowing for accumulated visits or transfers to other prisons for visits closer to home (Her Majesty's Prison & Probation Service, 2018 ). Family communication – additional information can be found in Table 14 .

External organisations

One review suggested that external voluntary agencies were not often contacted or referred to, despite their potential benefits in terms of costs and support for staff and PLiPWDs (du Toit et al., 2019 ). However, other papers reported that charities for PLiPWD, or older people, were involved in (or were recommended to be involved in): designing and/or delivering dementia training; being part of MDTs; informing the design of referral processes, screening, assessment and case finding tools; consulting on environmental design; creating and delivering social care plans (including running activity centres); advice and support; advocacy and; co-facilitating a cognitive stimulation therapy group (Alzheimer’s Society 2018 ; Brooke et al., 2018 ; Brown, 2014 , 2016 ; Correctional Investigator Canada, 2019 ; Department of Health, 2007 ; du Toit & Ng, 2022 ; du Toit et al., 2019 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Goulding, 2013 ; Her Majesty's Inspectorate of Prisons, 2014 ; HMP Hull, 2015 ; Her Majesty's Prison & Probation Service, 2018 ; Hodel & Sanchez, 2013 ; Moll, 2013 ; Peacock et al., 2018 ; Prisons and Probation Ombudsman, 2016 ; Sindano & Swapp, 2019 ; Tilsed, 2019 ; Treacy et al., 2019 ; Williams, 2014 ). It was also recommended that external organisations need to have a better knowledge and understanding of prisons and people living in prison, in order to better manage risk, and for clear information sharing protocols (du Toit & Ng, 2022 ).

(iv) Transfers

During the course of their sentence, people in prison may be transferred to other prisons for various reasons or to receive treatment in hospital. The need for MDT transfer plans to be developed was reported (Welsh Government and Ministry of Justice, 2011 ), as was the need to limit the number of prisoner transfers as moving accommodation is likely to have an adverse effect (Her Majesty's Prison & Probation Service, 2018 ; Patterson et al., 2016 ). It was recommended that transfers should take the distance from family and friends into account (Her Majesty's Prison & Probation Service, 2018 ), and that the ‘receiving’ facility (prison or healthcare setting) should be liaised with regarding health and social care, and risk (Her Majesty's Prison & Probation Service, 2018 ; Welsh Government and Ministry of Justice, 2011 ) to ensure continuity of care (Cipriani et al., 2017 ). A standard document transfer protocol was also postulated as useful, as documents need to be forwarded quickly as well (Brown, 2016 ; Tilsed, 2019 ; Welsh Government and Ministry of Justice, 2011 ). At the receiving facility, it was suggested that assessments and care plans should be reviewed on the day of the transfer (Brown, 2016 ; Her Majesty's Prison & Probation Service, 2018 ; National Institute for Health and Care Excellence, 2017 ; Welsh Government, 2014 ), and for re-inductions to be facilitated for prison transfers (Her Majesty's Prison & Probation Service, 2018 ).

(v) Release and resettlement

Most prisoners will be released from prison at the end of their sentence, although a number may die before their time is served. A number of areas were highlighted regarding the release and resettlement of PLiPWD, including the possibility of early release due to dementia.

Early release

A number of papers advocated for compassionate release policies and their actual use, or alternative custodial placements such as halfway houses or secure nursing homes, that would effectively result in the early release of PLiPWD (Brown, 2016 ; Cipriani et al., 2017 ; Correctional Investigator Canada, 2019 ; Dementia Action Alliance, 2017 ; Department of Health, 2007 ; du Toit & Ng, 2022 ; du Toit et al., 2019 ; Fazel et al., 2002 ; Gaston & Axford, 2018 ; Goulding, 2013 ; Her Majesty's Prison & Probation Service, 2018 ; Hodel & Sanchez, 2013 ; Inspector of Custodial Services, 2015 ; Maschi et al., 2012 ; Mistry & Muhammad, 2015 ; Pandey et al., 2021 ; Turner, 2018 ; Williams et al., 2012 ). Although, it has also been noted that early release may not be a popular idea for some sections of the community (du Toit et al., 2019 ; Garavito, 2020 ), it was also suggested that raising community awareness of dementia may ameliorate this (du Toit & Ng, 2022 ). It was reported that prisoners with dementia should be considered in any criteria set forth for early release, particularly given the high cost/low risk ratio which they represent (Baldwin & Leete, 2012 ; Correctional Investigator Canada, 2019 ; Department of Health, 2007 ; Goulding, 2013 ; Her Majesty's Prison & Probation Service, 2018 ; Inspector of Custodial Services, 2015 ; Maschi et al., 2012 ; Murray, 2004 ; Williams et al., 2012 ). For prisoners who do not understand the aims of prison, continuing to hold them may be a contravention of human rights and equality laws – particularly where health and social care is inadequate (Baldwin & Leete, 2012 ; Dementia Action Alliance, 2017 ; Fazel et al., 2002 ; Gaston & Axford, 2018 ; Murray, 2004 ). It was also emphasised that the existence of units and programmes for PLiPWD should not be used to legitimise prison as an appropriate place for PLiPWD (Correctional Investigator Canada, 2019 ). More information can be found in Table 15 .

Resettlement

Ten different areas were identified in the literature which related to the issues PLiPWD leaving prison may face on their release and resettlement into the community, these were:

(a) In-prison release preparation

Specific pre-release programmes or services for older people or those living with dementia may be required (Department of Health, 2007 ; Williams et al., 2012 ), with prisoners being cognitively screened prior to release (Goulding, 2013 ), although the latter was only found in 10% of prisons in one study (Forsyth et al., 2020 ). Other suggestions for programme content included: self-efficacy, health, staving off dementia and associated anxiety, accessing services, addressing institutionalisation, setting up email addresses, and the provision of information packs on national, regional and local services and resources (Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Williams et al., 2012 ).

It has been suggested that release plans and transitions be facilitated by an MDT including prisoners, the voluntary sector, offender managers, and other appropriate community-based organisations (du Toit et al., 2019 ; Feczko, 2014 ; Goulding, 2013 ; Her Majesty's Prison & Probation Service, 2018 ; Inspector of Custodial Services, 2015 ; Moll, 2013 ; Welsh Government and Ministry of Justice, 2011 ). Recommended plan content included: risk management strategies, health, social care, housing, finance, employment, leisure and voluntary sector considerations (Welsh Government and Ministry of Justice, 2011 ). It was also suggested that Circles of Support and Accountability (CoSA), primarily associated with sex offenders, could be set up for PLiPWD as a means to support those leaving prison and settling back into the community particularly without family support (Her Majesty's Prison & Probation Service, 2018 ).

Challenges to release preparation were identified as: a lack of resources, (Turner, 2018 ) the lack of clarity regarding staff resettlement roles (Inspector of Custodial Services, 2015 ), and the lack of resettlement provision offered at sex offender prisons in England and Wales (Her Majesty's Prison & Probation Service, 2018 ).

A number of papers reported the key role that family and friends can or do play in supporting PLiPWD leaving prison, and that this should be supported or facilitated by prison staff (Brown, 2016 ; Her Majesty's Prison & Probation Service, 2018 ; Goulding, 2013 ). Initially this could include encouraging diagnosis disclosure (Dillon et al., 2019 ), using prison leave to maintain relationships (Her Majesty's Prison & Probation Service, 2018 ), involvement in discharge planning (Welsh Government and Ministry of Justice, 2011 ), and placing prison leavers close to family upon release and ensuring family are supported (Correctional Investigator Canada, 2019 ; Gaston & Axford, 2018 ). Where PLiPWD lack family, setting up CoSAs as described above may be useful (Her Majesty's Prison & Probation Service, 2018 ).

(c) Probation

It was suggested that probation staff should have training to work with older people, and that some offender managers could specialise in this work (Department of Health, 2007 ; Welsh Government and Ministry of Justice, 2011 ). Probation officers or offender managers are or can be involved in resettlement planning, (Her Majesty's Prison & Probation Service, 2018 ; Welsh Government and Ministry of Justice, 2011 ), arranging accommodation (Inspector of Custodial Services, 2015 ), liaising with agencies such as health care or social services, checking that PLiPWD are accessing these services and disseminating reports of to-be released prisoners to relevant parties (Department of Health, 2007 ; Moll, 2013 ; Welsh Government and Ministry of Justice, 2011 ). Importantly, the forwarding of important documents to offender managers by the prison should be routine (Department of Health, 2007 ; Moll, 2013 ). It was also recommended that probation staff should visit people in prison before release if they live out of area (Department of Health, 2007 ). The work of probation services was reportedly hampered by limited resources (Brown, 2016 ).

Continuity of care upon release can be difficult, and it was suggested that it could be a role of prison healthcare to ensure this (including registering with the local GP and dentist (Cipriani et al., 2017 ; Department of Health, 2007 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Her Majesty's Prison & Probation Service, 2018 ; Inspector of Custodial Services, 2015 ; Welsh Government and Ministry of Justice, 2011 ). There appeared to be some differences regarding the distribution of full healthcare reports to offender managers and other appropriate agencies with some prisons sending them, some only if requested, and some not providing them on grounds of confidentiality (Moll, 2013 ). Typically, it was recommended that it was better for to-be released older prisoners if these reports were disseminated (Department of Health, 2007 ). It was also suggested that healthcare staff in prison and from the community form part of multi-disciplinary release planning, and that these plans include health considerations and healthcare staff advice on issues of accommodation (du Toit & Ng, 2022 ; Inspector of Custodial Services, 2015 ; Moll, 2013 ; Welsh Government and Ministry of Justice, 2011 ).

(e) Social care

Some papers reported that social workers can and should be involved in the process of resettlement (Department of Health, 2007 ; Welsh Government and Ministry of Justice, 2011 ) and release preparation (Goulding, 2013 ). Continuity of social care arranged with the local authority was also recommended (Her Majesty's Prison & Probation Service, 2018 ; Welsh Government and Ministry of Justice, 2011 ).

(f) Accommodation

Release planning should include plans for accommodation, and involve housing agencies or care services in the community in that planning (Welsh Government and Ministry of Justice, 2011 ). Importantly, people in prison may need help in registering for housing, and their homes may be in need of adaptation in response to their health or social care needs (Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ). Nursing homes and other care providing facilities were reported to be reluctant to accommodate people who have been in prison (Brown, 2014 ; Brown, 2016 ; Booth, 2016 ; Correctional Investigator Canada, 2019 ; du Toit et al., 2019 ; Gaston, 2018 ; Garavito, 2020 ; Goulding, 2013 ; Inspector of Custodial Services, 2015 ). This was described as particularly the case for those who were living with dementia (Brown, 2014 ; Correctional Investigator Canada, 2019 ; Dillon et al., 2019 ), with further issues reported in accommodating those who have committed sex offences (Brown, 2014 , 2016 ; Dillon et al., 2019 ; Garavito, 2020 ; Inspector of Custodial Services, 2015 ). Concerns regarding the safety of other residents and the views of their families, and the rights of victims in general, were cited as reasons behind these placement difficulties (Brown, 2014 ; Goulding, 2013 ) – one paper reported that there had been community protests (Brown, 2016 ).

It was suggested that prisons need to build better relationships with care providers in the community, which had reportedly been forged by some (Brown, 2016 ; Goulding, 2013 ; Inspector of Custodial Services, 2015 ), and that they could also provide education and support to these services (Booth, 2016 ). However, it was also noted that there may be a need for specialist residential units to be created in the community for people released from prison with dementia (Inspector of Custodial Services, 2015 ), with an example of a state-run facility for ex-prisoners in the United States (Goulding, 2013 ), and particular attention for younger ex-prisoners with dementia (Brown, 2014 ). A number of papers reported that if accommodation could not be arranged for people, this largely resulted in them remaining in prison until it was (Correctional Investigator Canada, 2019 ; Goulding, 2013 ; Inspector of Custodial Services, 2015 ; Peacock et al., 2018 ; Soones et al., 2014 ).

(g) Finance

Imprisonment likely leads to a loss of income, meaning that older prisoners who may have served more lengthy sentences are likely to be poorer, particularly if unable to work in prison (Baldwin & Leete, 2012 ; Gaston, 2018 ). Therefore, it was suggested that release planning ought to include issues of finance (Welsh Government and Ministry of Justice, 2011 ). Given that it has been suggested that people in prison should be given advice on pensions and welfare benefits, and help to arrange these (Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Goulding, 2013 ), addressing this would seem to be an area of particular use for older people leaving prison who may have additional problems in these areas, and for those who may need assistance in arranging their financial affairs because of their deteriorating health problems.

(h) Employment and education

People’s employment prospects are likely to be impacted upon release from prison, particularly for older people who may have served long sentences (Gaston, 2018 ). Where appropriate, it was recommended that release planning should include issues around employment (Welsh Government and Ministry of Justice, 2011 ), that information packs for people should include sections on education and employment, and that it could be useful to help people make links with the Department for Work and Pensions (Her Majesty's Prison & Probation Service, 2018 ).

(i) Leisure

Leisure activities and resources could be considered in release planning, and included in pre-release information packs for prisoners (Her Majesty's Prison & Probation Service, 2018 ; Welsh Government and Ministry of Justice, 2011 ).

(j) Charities and voluntary sector organisations

It was recommended in a number of papers that charity and voluntary sector organisations working with PLiPWD be involved in release planning (Department of Health, 2007 ; du Toit et al., 2019 ; Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ; Welsh Government and Ministry of Justice, 2011 ), continuity of care (Moll, 2013 ), and in providing support during the transition and after (du Toit & Ng, 2022 ; Welsh Government and Ministry of Justice, 2011 ). It was also suggested that in general it would be useful for PLiPWD to have contact with these organisations (Department of Health, 2007 ; Her Majesty's Prison & Probation Service, 2018 ; Inspector of Custodial Services, 2015 ), and that they may be well-placed to develop information packs for prisoners on release regarding local amenities, services and resources (Her Majesty's Prison & Probation Service, 2018 ).

(vi) Cross-cutting themes

Eight more generalised concerns were also described which had a clear impact on the passage of PLiPWD through prison, on release and resettlement in the community, and on the issues raised thus far in the review.

Principles-philosophy

The principles suggested to underpin the support of PLiPWD are that it should be person-centred, holistic, adhere to human rights and dignity principles, proactive, health promoting, and enabling – making choices but supported if needed (Brown, 2014 , 2016 ; Christodoulou, 2012 ; Cipriani et al., 2017 ; Correctional Investigator Canada, 2019 ; Department of Health, 2007 ; Dillon et al., 2019 ; du Toit & Ng, 2022 ; Gaston & Axford, 2018 ; Her Majesty's Inspectorate of Prisons, 2017b ; Her Majesty's Prison & Probation Service, 2018 ; Mackay, 2015 ; Maschi et al., 2012 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ; Wilson & Barboza, 2010 ). Conversely, clashes in philosophies between prison staff, and health and social care staff have been reported with security trumping care in many cases, which can have a negative impact (du Toit & Ng, 2022 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Goulding, 2013 ; Mackay, 2015 ; Murray, 2004 ; Patterson et al., 2016 ; Prisons and Probation Ombudsman, 2016 ; Treacy et al., 2019 ; Williams, 2014 ). It was suggested that positioning dementia as more than just a health issue and fostering a whole-prison care-custody model or approach, with clearly defined roles for ‘care’ and ‘custody’, may be useful in resolving this (du Toit & Ng, 2022 ; Public Health England, 2017b ; Welsh Government and Ministry of Justice, 2011 ).

A number of papers ( n  = 15) reported that budget and resource limitations had a variety of negative impacts including difficulties in providing: appropriate assessment, support and accommodation to PLiPWD; specialist accommodations, plans for which were then curtailed; delivering programmes and activities; healthcare cover; and, staff training (Booth, 2016 ; Christodoulou, 2012 ; Correctional Investigator Canada, 2019 ; Dementia Action Alliance, 2017 ; Dillon et al., 2019 ; du Toit et al., 2019 ; du Toit & Ng, 2022 ; Goulding, 2013 ; HMP Hull, 2015 ; Jennings, 2009 ; Mackay, 2015 ; Moll, 2013 ; Moore & Burtonwood, 2019 ; Pandey et al., 2021 ; Patterson et al., 2016 ; Peacock et al., 2018 ; Treacy et al., 2019 ; Turner, 2018 ). Ultimately, lack of resources has reportedly led to a system that is not able to cope appropriately with PLiPWD (Moll, 2013 ; Williams et al., 2012 ; Wilson & Barboza, 2010 ), with associated problems transferring out of the prison system into probation and care systems when people are released (Williams et al., 2012 ).

It has been suggested that PLiPWD in prison should be treated as if they have capacity to make decisions such as giving or withholding consent for treatment, unless it is proven otherwise. This is consistent with legislation such as the Mental Capacity Act (Prisons and Probation Ombudsman, 2016 ). It has been recommended that healthcare staff should conduct capacity assessments if there are concerns (National Institute for Health and Care Excellence, 2017 ; Welsh Government and Ministry of Justice, 2011 ), and be trained to do so (Maschi et al., 2012 ; Welsh Government, 2014 ). It is of note that an ombudsman report showed that PLiPWD who died lacked access to mental capacity assessments (Peacock et al., 2018 ). For PLiPWD, who are likely to lack capacity as their condition progresses, early education about, and development of, advance directives has been advocated (Brown, 2016 ; Cipriani et al., 2017 ; Inspector of Custodial Services, 2015 ; Maschi et al., 2012 ; Prisons and Probation Ombudsman, 2016 ), and staff should be trained on this (Maschi et al., 2012 ). It has also been suggested that family members, independent mental capacity advocates or healthcare proxies could or should be used for PLiPWD who lack capacity in making care, welfare and financial decisions (Brown, 2016 ; Soones et al., 2014 ), supported by legislation and oversight, as opposed to prison or healthcare staff making decisions (Correctional Investigator Canada, 2019 ).

The issue of ‘risk’ related to PLiPWD revolves around four areas: (i) assessment, (ii) management, (iii) disciplinary procedures, and (iv) safeguarding. Full details can be found in Table 16 .

There were a number of additional facets to risk concerns regarding PLiPWD described in the papers. There were concerns that the lack of understanding of the impact of dementia on people’s behaviour could ultimately lead to people being held in prison for longer periods on account of seemingly transgressive or aggressive behaviour that could in fact be related to their dementia difficulties (Dementia Action Alliance, 2017 ; Mistry & Muhammad, 2015 ; Treacy et al., 2019 ). In one study, a prisoner with dementia was transferred to another prison because staff felt that they were ‘grooming’ an officer (Treacy et al., 2019 ), likely lengthening their overall prison stay. There was also a recurring issue in fatal incidents investigations in England and Wales of prisoners being restrained whilst dying in hospital, a practice described as unnecessary in light of their likely frail state (Peacock et al., 2018 ; Prisons and Probation Ombudsman, 2016 ). One paper suggested linking future accommodation options and considerations for Release on Temporary Licence to a PLiPWD’s risk of reoffending, as well as the severity of their symptoms (Forsyth et al., 2020 ). Moore and Burtonwood ( 2019 ) also observed that a lack of risk assessment protocols was a barrier to release of PLiPWD., and as Table 16 suggests, a comprehensive risk assessment, applied by appropriately trained staff should make health and its impact on future offending more salient to aid this.

There were recommendations that PLiPWD should have the opportunity to make choices in their treatment and care. This included input into care plans or making informed decisions about their care (Department of Health, 2007 ; du Toit & Ng, 2022 ; National Institute for Health and Care Excellence, 2017 ; Welsh Government and Ministry of Justice, 2011 ), as well as developing advance directives particularly early in a person’s sentence (Brown, 2016 ; Cipriani et al., 2017 ; Inspector of Custodial Services, 2015 ; Maschi et al., 2012 ; Pandey et al., 2021 ; Peacock et al., 2019 ; Prisons and Probation Ombudsman, 2016 ), and choosing ‘preferred’ places to die (Her Majesty's Prison & Probation Service, 2018 ).

Protected characteristics

There was a reported need for culturally appropriate assessments, treatment and activities (Brooke et al., 2018 ; Department of Health, 2007 ; Hamada, 2015 ; Welsh Government and Ministry of Justice, 2011 ), spiritual support (Welsh Government and Ministry of Justice, 2011 ), multilingual information (Welsh Government and Ministry of Justice, 2011 ), and the recognition of gender differences in dementia healthcare needs (Brown, 2014 ; Department of Health, 2007 ; Williams et al., 2012 ). It was also highlighted that racism makes the experience of living with dementia in prison more problematic (Brooke et al., 2018 ; Brown, 2014 ; Correctional Investigator Canada, 2019 ). There were some examples of policy and practice within prisons which considered some protected characteristics: assessment tools in different languages (Patterson et al., 2016 ), additional support for PLiPWD to plan care (Department of Health, 2007 ; Welsh Government and Ministry of Justice, 2011 ), and the development of culturally appropriate care planning (Hamada, 2015 ). Hamada ( 2015 ) also advocated assessment and treatment that was culturally ‘competent’ and respectful, and which acknowledged the importance of culture and diversity.

An overall need to tackle dementia- and age-related stigma was also reported in some papers, and the need to foster cultures that are age-respectful should be reflected in staff training (Department of Health, 2007 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ), In addition, practices which openly discriminate such as the lack of: dedicated dementia resources (Turner, 2018 ), appropriate lower category prison places (Department of Health, 2007 ; Welsh Government and Ministry of Justice, 2011 ), and appropriate accommodation on release, which at times prevents release, should also be challenged (Correctional Investigator Canada, 2019 ; Forsyth et al., 2020 ; Ministry of Justice, 2013 ; Prisons and Probation Ombudsman, 2016 ). There was also a lack of research into the interaction between protected characteristics and dementia in prison (Brooke & Jackson, 2019 ; Treacy et al., 2019 ; Williams et al., 2012 ).

Collaboration

Many papers advocated the need for prisons and specialist dementia units to adopt a collaborative MDT approach drawing from staff teams across the prison regarding: the identification and support of prisoners with dementia, care planning, the disciplinary process, the development, dissemination and implementation of policy, and in environmental change and the building of new prisons (Brooke et al., 2018 ; Brown, 2014 , 2016 ; Christodoulou, 2012 ; Cipriani et al., 2017 ; Dillon et al., 2019 ; Department of Health, 2007 ; Feczko, 2014 ; Forsyth et al., 2020 ; Gaston & Axford, 2018 ; Her Majesty's Inspectorate of Prisons, 2014 , 2016 ; HMP Hull, 2015 ; HMP Littlehey, 2016 ; Her Majesty's Prison & Probation Service, 2018 ; Inspector of Custodial Services, 2015 ; Moll, 2013 ; Patterson et al., 2016 ; Peacock et al., 2018 ; Peacock, 2019 ; Prisons and Probation Ombudsman, 2016 ; Sindano & Swapp, 2019 ; The King’s Fund 2013 ; Tilsed, 2019 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 , 2014 ; Williams, 2014 ). There were examples of prisoners collaborating with staff in the care of PLiPWD as peer supporters, and having joint staff-prisoner supervision and training (Brooke & Jackson, 2019 ), of joint staff-prisoner wing meetings in one prison (Treacy et al., 2019 ), and of the co-designing of services and activities in others (Her Majesty's Prison & Probation Service, 2018 ; Treacy et al., 2019 ). It was suggested that this collaborative way of working should be supported by an information sharing protocol, clear definitions of staff and peer supporter roles and responsibilities, and training (Brooke & Jackson, 2019 ; Dillon et al., 2019 ; du Toit & Ng, 2022 ; HMP Littlehey, 2016 ; Turner, 2018 ). It was reported that there had been a lack of communication and coordination of this process in some prisons which had a negative impact on all involved (Brooke & Rybacka, 2020 ; Forsyth et al., 2020 ; Moll, 2013 ; Prisons and Probation Ombudsman, 2016 ).

It was also suggested that the prisons collaborate with healthcare, hospice and dementia specialists in the community and with external charitable organisations (Brooke et al., 2018 ; Brown, 2014 ; Cipriani et al., 2017 ; du Toit & Ng, 2022 ; Gaston, 2018 ; Gaston & Axford, 2018 ; Goulding, 2013 ; HMP Hull, 2015 ; HMP Littlehey, 2016 ; Her Majesty's Prison & Probation Service, 2018 ; Moll, 2013 ; Peacock, 2019 ; Prisons and Probation Ombudsman, 2016 ; Sindano & Swapp, 2019 ; Tilsed, 2019 ; Treacy et al., 2019 ; Welsh Government and Ministry of Justice, 2011 ; Williams, 2014 ). In addition, inter-prison networks were recommended to be developed to share good practice across prisons (Dementia Action Alliance, 2017 ; Moll, 2013 ; Peacock et al., 2019 ; Prisons and Probation Ombudsman, 2016 ).

Information-sharing

A number of papers ( n  = 7) recommended the need for a clear information sharing protocol regarding the assessment and support of PLiPWD (Brooke et al., 2018 ; Dillon et al., 2019 ; Department of Health, 2007 ; Goulding, 2013 ; Moll, 2013 ; Tilsed, 2019 ; Welsh Government and Ministry of Justice, 2011 ), or a register (Forsyth et al., 2020 ). Particular attention to the interface between healthcare and prison staff and peer supporters was suggested, where it has been reported that privacy regulations have sometimes prevented contributions to collateral histories (Feczko, 2014 ) and the sharing of care plans, impairing their ability to offer appropriate support (Inspector of Custodial Services, 2015 ). Also, it may be against the wishes of the person with dementia, and informed consent should be sought (Forsyth et al., 2020 ; Moll, 2013 ). This lack of information can have a detrimental effect on a person’s health and wellbeing (Brown, 2014 , 2016 ; Feczko, 2014 ; Inspector of Custodial Services, 2015 ), and so discussion of this was highlighted as important, particularly where the safety of the person or others were concerned (National Institute for Health and Care Excellence, 2017 ). A care plan which gives only very basic information to staff and peer supporters was used in a couple of prisons (Goulding, 2013 ; Williams, 2014 ).

There also appeared to be variance with respect to whether healthcare staff disclose a dementia diagnosis to the person diagnosed with dementia. A couple of prisons’ policy was to share a diagnosis and involve family in doing so (Maschi et al., 2012 ; Welsh Government and Ministry of Justice, 2011 ; Wilson & Barboza, 2010 ), however, in one prison disclosed if a person was judged to be able to cope with it, and another only disclosed if asked (Brown, 2016 ). The importance of disclosure to family allowing them to contribute to assessments, planning and support was also emphasised in some papers (Brown, 2016 ; Dillon et al., 2019 ; National Institute for Health and Care Excellence, 2017 ; Welsh Government and Ministry of Justice, 2011 ).

This review has explored the literature regarding all parts of the custodial process and its impact on people living in prison with cognitive impairment and dementia, which includes: reception, assessment, allocation, training, policy, healthcare, accommodation, adaptation, routine, access to family and external agencies, transfer and resettlement. We found evidence that problems had been identified in each of these parts of the process. We also identified a number of cross-cutting themes which interacted with the issues identified across the prison journey including: principles or philosophy regarding care; capacity; resources; considerations of risk; scope for choice; peoples’ protected characteristics; collaboration; and, information sharing. Broadly, our findings were similar to those found in previous reviews, regarding the problems with the prison process identified, and the lack of robust outcomes, and policy guidance regarding PLiPWD (Brooke and Rybacka, 2020 ; Peacock et al., 2019 ).

The aim of this review was to identify areas of good practice and for recommendations that could inform the development of prison dementia care pathways. There is a considerable breadth to the findings, but the main recommendations that have arisen from the review are:

To screen prisoners for cognitive difficulties at reception, from either 50 or 55 years

An initial older-person specific health and social care assessment, post-screening – from either 50 or 55 years, and repeated (from 3 – 12 months)

A spectrum of healthcare to be delivered including preventative, long-term and palliative care, with continuity of care upon release, and in tandem with social care

Mixed views about appropriate accommodation, but it needs to run along a continuum from independent living to 24-h care, with decisions possibly made after health assessments

Environments need to be made more older-person or dementia friendly, using checklists available, and with the voluntary sector as potential partners

A need for prison staff training on dementia, and further training for healthcare staff

The use of peer supporters was broadly reported positively, and were seemingly frequently used. However, there needs to be adequate training and support, and not to be used to do the work that is the statutory duty of health and social care staff

Equal access to activities and services, especially programmes which help people move through the system (such as offending behaviour), as well as opportunities to earn additional monies, and that provide structure and routine on wings

The maintenance of family links, and for families to be supported, are important for PLiPWD, and may be particularly so on release and resettlement

Prisons may also need to work with external care agencies to ensure placements upon release, or alternative specialist care facilities may need to be created

The main barriers to implementing these recommendations are a lack of policy or guidance at local, regional and national levels to support staff in working with PLiPWD, and also the lack of budget and resources available. The latter would also include infrastructure issues, such that a number of prisons are not appropriate for people living with dementia, and could be expensive to modify to become so, coupled with a lack of currently available alternative facilities for PLiPWD to be released to in the community. The lack of use of compassionate release is also an issue here, including during the COVID-19 pandemic, with only 54 people released (Halliday & Hewson, 2022 ). Lastly, the roles that each professional and peer group had regarding PLiPWD needed clarification in some prisons, including some resolution of the ‘clash’ of philosophies (control v care) underpinning this.

In terms of ‘solutions’, multiple organisations have advocated for years for the need for national policy to assist prisons with older people in prison, including those living with dementia (Cornish et al., 2016 ; HM Inspectorate of Prisons, 2004 , 2019 ; Prisons & Probation Ombudsman, 2016 , 2017 ). This was eventually accepted and commissioned by the UK government, although it has not been released as yet (Justice Committee, 2020 ). It has also been suggested that at a more local level, existing policies could be adapted to be more appropriate for PLiPWD – such as restraint policies for frail prisoners, and disciplinary procedures which reflect the impact that dementia may have on behaviour (Department of Health, 2007 ; Treacy et al., 2019 ). Considerations around capacity and consent would need to be weaved in, as well as a focus on the intersection with other protected characteristics. These adaptations would also need to extend to services and activities to ensure that people have equal access and opportunities. A number of reports highlighted the contribution that greater collaboration with partners in external health and social care teams could have, as well as partnerships with the voluntary sector. These could potentially assist in multiple areas including training staff and peer supporters, providing activities, assisting release preparation, at a relatively low cost, to high benefit. There were some recommendations that prisons adopt a whole-prison approach to dementia that focuses on being person-centred, health and human rights focused that may help to ameliorate some differences in philosophical approach between various staff and peer groups in prisons.

A number of potential areas for future research were also indicated by the literature, which would also support the development of prison pathways. These would include: (i) induction to prison, and (ii) release and resettlement from prison, which are important beginning and end-points, but which are under-researched; (iii) the validation of a screening tool for use in prisons, and the development or adaptation of prison-specific health and social care assessments; (iv) the interaction of protected characteristics and dementia, and the need for more culturally and gender aware pathways; (v) the paucity of research conducted in low and middle-income countries, that needs to be addressed; (vi) dementia and age-related stigma in prisons; and (vii) evaluations of all elements of the prison pathway for PLiPWD to undertaken including training, the role of peer supporters, and targeted programmes.

Strengths and limitations of the review

One key strength of this review is its comprehensiveness, particularly as it includes much grey literature. Given the lack of robust evaluation in this area, it was felt that this was necessary to represent the volume of work that has nonetheless taken place. There are, however, a number of limitations of this review. Firstly, despite the use of broad search terms, there may be the possibility that some relevant research was missed, either because of deficiencies in our searches or because of publication bias. Additionally, whilst there are twenty-two guidance and inspection documents included in this review, it is possible that some grey literature might also remain unidentified, particularly outside of the UK where the review was undertaken. Secondly, this review may be subject to a selection bias, as the yielded search results might have included literature that were excluded but which may have indirectly impacted upon the care pathways elements explored in the review. There is also a language bias, and whilst this may reflect the languages spoken by the review team members, it is also reflective of the “northern epistemic hegemony” (Aas, 2012 ), that also may have resulted in the review being largely populated by papers from high income countries. Thirdly, no formal assessment of study quality was undertaken. This is in keeping with scoping review methodology which focuses on breadth, but is nonetheless an important shortcoming inherent in scoping reviews more generally (Arksey & O’Malley, 2005 ).

We have completed the most comprehensive review of the literature on PLiPWD in prisons to date that we have found, including a synthesis of the extensive grey literature, and found important gaps in the literature. Our review includes a mixture of academic research, policy and position papers which identified an increasing number of prisoners with dementia or cognitive impairment as an issue, but there were more limited descriptions of what should be done, and even less describing implementation of these. Most of the literature came from developed nations where extensive assessment and care services are in place for PWD in the community, although a key question is whether prison populations are given easy access to these existing services or whether bespoke services for prisoners are required. We suggest this literature now needs to be drawn together to inform interventions for PLiPWD in the criminal justice system which can be piloted and evaluated, and inform the development of robust dementia care pathways for prisons.

Availability of data and materials

All data and materials used in this review are included in this article and its appendices.

There is no standard cut-off age for older people living in prison, but it is typically set at least ten to fifteen years lower than the general population. People in prison are thought to age more rapidly due to both pre- and post-imprisonment chaotic lifestyles, substance misuse and less healthcare access and use, as well as the ‘pains of imprisonment’. See Williams et al., ( 2012 ) for further discussion.

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Acknowledgements

We would like to thank all the funders for their contributions towards this review. We also would like to thank the key stakeholders, especially the prison advisors and old age psychiatry and care advisors, who contributed towards shaping and contextualising this evidence review.

This is a summary of research which was partly funded by the National Institute for Health Research (NIHR) Applied Research Collaboration East of England - previously, the Collaboration for Leadership in Applied Health Research and Care East of England – and the Cambridgeshire and Peterborough NHS Foundation Trust (CPFT), as part of the wider prison care programme. The views expressed are those of the author(s).

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ST was the lead researcher and conceptualised, designed, searched, analysed and interpreted data, and led on writing the manuscript. VP provided crucial and extensive library support. SM and NS were involved in screening and extracting data as well as analysis (SM), reviewing and editing various versions of the manuscript. TVB was the Principle Investigator/Study Lead and contributed towards conceptualisation, design, data quality control, manuscript reviewing and editing, and supervising all aspects of the study. BRU was Co-Principle Investigator and provided clinical advice input. ST, SM and TVB revised the peer-reviewed manuscript. All authors read and approved the final submitted manuscript.

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Treacy, S., Martin, S., Samarutilake, N. et al. Dementia care pathways in prisons – a comprehensive scoping review. Health Justice 12 , 2 (2024). https://doi.org/10.1186/s40352-023-00252-7

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Surgical and radiological perspectives for the spinal accessory nerve passing through a fenestrated internal jugular vein: case series and literature review

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Pierre Guarino, Paolo Tesauro, Leone Giordano, Claudio Donadio Caporale, Livio Presutti, Francesco Mattioli, Surgical and radiological perspectives for the spinal accessory nerve passing through a fenestrated internal jugular vein: case series and literature review, Journal of Surgical Case Reports , Volume 2024, Issue 4, April 2024, rjae099, https://doi.org/10.1093/jscr/rjae099

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The preservation of the spinal accessory nerve represents a key goal in head and neck oncologic surgery during selective neck dissection. This study aims to illustrate the anatomical variants of the XI cranial nerve, delving into the relationship between the spinal nerve and the internal jugular vein, as well as the surgical implications. Two cases of patients who underwent oncologic surgery with neck dissection are described. Both cases found the spinal accessory nerve passing through the fenestration of the internal jugular vein. Alongside this case series, an independent literature review was conducted using the Medline and PubMed databases. In the majority of cases (67% – 96%), the spinal accessory nerve traces a lateral course to the internal jugular vein. Less frequently, the XI cranial nerve courses medial to the internal jugular vein. More rarely, as described in this case series, the nerve crosses through the fenestration of the vein (0.48% – 3.3%). 

During neck dissection, the spinal accessory nerve (SAN) is encountered on Level II at the level of the posterior belly of the digastric muscle where it crosses laterally and anterior the internal jugular vein (IJV). A medial course of the nerve is reported in literature with an incidence that varies from work to work. Rarely, the SAN can pass through a fenestration of the IJV or can split around the IJV [ 1 , 2 ]. Nowadays, preservation of the SAN is the standard of care even though iatrogenic injury to the SAN and to the IJV still occurs. In order to reduce the risk of iatrogenic injury a detailed study of the radiological anatomy together with an extensive knowledge of the anatomical variants of the SAN and of the IJV represent the surgeon’s most important tools [ 3–6 ].

A 78-year-old man with a follow-up PET for a Type B mantle cell lymphoma finding of uptake in in the epiglottis and left laterocervical region is reported here. The patient was diagnosed with squamous cell carcinoma (SCC) after fine needle aspiration of an adenopathy at Level II. His workup included an MRI of the neck with contrast enhancement, which revealed laterocervical lymphadenopathy (bilateral level IIa and left Level III with the biggest adenopathy with maximum diameter of 12 × 12 mm) and thickening of the epiglottis extended at the floor of the left vallecula. No mention was made of an IJV abnormality in the report even though it was noticed during the preoperative evaluation by the surgeon ( Fig. 1 ). The fibroendoscopic evaluation of the neck showed an asymmetry of the epiglottis with the presence of vegetating tissue at the level of the left lingual border of the epiglottis, preserved motility of the vocal cords, and normal breathing space. The patient’s clinical stadiation was cT2 N2c M0. The multidisciplinary meeting indicated an endoscopic partial laryngectomy and bilateral laterocervical dissection. The cervical lymph node dissection on the left side discovered a duplication of the IJV which resulted dilated in comparison to the IJV on the right side. The SAN passed medially to the anterior vein and laterally to the posterior vein. Both the IJV and the SAN were preserved during the procedure ( Fig. 2 ).

MRI showing the fenestration of the IJV; left image: sagittal section showing the fenestration (arrowhead) of the left IJV; right image: axial section showing the anterior (dotted arrowhead) and the posterior (arrowhead) part of the fenestrated IJV.

SAN passes through medially to the anterior part (filled triangle) and laterally to the posterior part (star) of the fenestrated IJV (arrowhead); ICA: internal carotid artery; SCM: sternocleidomastoid muscle.

A 72-year-old woman underwent total laryngectomy + thyroid isthmectomy + bilateral (II–IV) laterocervical dissection for a G3 SCC of the right hemilarynx (involving the right true and false vocal cord and extending to the anterior commissure and left vocal cord). The preoperative radiology report did not mention any IJV variations. Nonetheless, a left IJV fenestration was observed in the preoperative study, raising the suspect of a possible SAN variant ( Fig. 3 ). Pathological stadiation of the patient was pT4a (infiltration of thyroid cartilage and prethyroid soft tissue) pN2c, cM0. Intraoperatively, the fenestration of the left IJV, that was noticed during the CT, was confirmed. As for the first patient presented, the SAN nerve passed medially to the anterior vein and laterally to the posterior vein ( Fig. 4 ).

CT scan showing the fenestration of the IJV; left image: sagittal section showing the fenestration (arrowhead) of the left IJV; central image: coronal section showing the anterior (dotted arrowhead) and the posterior (arrowhead) part of the fenestrated IJV; right image: axial section showing the anterior (dotted arrowhead) and the posterior (arrowhead) part of the fenestrated IJV.

SAN passes through medially to the anterior part (filled triangle) and laterally to the posterior part (star) of the fenestrated IJV (arrowhead); CCA: common carotid artery.

An in-depth understanding of the anatomy of the lateral neck is essential to avoid injury to the IJV during SAN dissection. A literature review of the anatomic relationship of the SAN to the IJV is presented in Table 1 .

Literature review of the incidence of the anatomical variants of the SAN.

As shown, Krause, Soo, and Kierner conducted cadaveric studies [ 7–9 ]. Soo and Kierner reported an equal frequency of the lateral (56%) and medial (44%) position of the nerve compared to the IJV, while Krause reported an incidence of 72.5% of the nerve crossing lateral to the IJV. Krause also observed one case of SAN “fenestrating” through the IJV. Saman’s cadaveric study supported Krause’s data, reporting a 79.8% of SAN coursing lateral to the IJV, 19% of SAN medial to the IJV, and 1.2% of SAN “fenestrating” through the IJV [ 10 ].

In vivo studies reported a higher frequencies of the nerve crossing lateral to the IJV. Levy et al. [ 11 ], Hinsley et al. [ 12 ] and Taylor et al. [ 13 ] showed the SAN lateral to the IJV in 99.25%, 96.6%, 95.7% and the SAN medial to the IJV in 0.75%, 2.6% and 2.8%. Taylor and Hinsley also reported <1% of cases of SAN “fenestrating” to the IJV; moreover, Taylor described a new anatomical variant of the SAN in which the nerve divides and travels both medial and lateral to the IJV.

The higher incidence of the nerve crossing lateral to the IJV in in vivo studies compared to cadaveric is explained by Hinsley and Taylor as follows. The IJV of cadavers can collapse, determining a higher incidence of the medial position of the nerve in relation to the IJV. Moreover, in vivo studies which are conducted on oncological patients focus on the relation between the nerve and IJV at the level of the posterior belly of the digastric muscle, while cadaveric studies do not always keep the same reference points.

In 2009, Lee published an in vivo study in which he found a surprising 57.4% incidence of SAN medial to the IJV. However, these results have not been confirmed by other studies [ 14 ].

In his in vivo study, Lee pointed out that the variation of the course of the SAN correlates with a variation in the number of lymph node of the Levels IIa and IIb. Therefore, a lateral course of the SAN increases the Level IIb area, and subsequently, the number of lymph nodes.

Regarding the rarer anatomic variants of the SAN, Table 1 shows an incidence of the SAN “fenestrating” the IJV ranging from 0.8% to 2.2% [ 15–17 ]. Taylor also describes a new anatomical variant of the spinal nerve splitting around the IJV. However, there are no other reports of this variant of the SAN and no iconographic material results available.

So what tools can a surgeon use to uncover a rare anatomical variant of the SAN? In 2012, Hashimoto published a case report where they were able to identify the fenestration of the IJV with a contrast-enhanced CT. However, Ozturk warns us that even though the fenestration of the IJV is strongly associated with the SAN traveling through the fenestration, this is not to be taken for granted as it can also travel medial or lateral to the fenestration [ 18 ].

In both cases presented in this article, the surgeon noticed the fenestration of the IJV during the preoperative imaging which was not indicated in the radiologist report. As the radiologist priority concerns the extension of the tumor and lymph nodal spread, it falls to the surgeon to enquire about potential anatomical variants of the IJV during the scan.

The literature review conducted in this study confirms that the SAN crosses lateral to the IJV in the majority of cases. However, incidental finding of variants of the SAN, as presented in the case reports, can occur intraoperatively, resulting in iatrogenic lesion of the SAN or of the IJV. Therefore, an upfront radiological study together with a deep knowledge of the possible variants of the SAN are the surgeon’s keystones to reduce the cases of damage to the SAN or to the IJV. The authors also hope that this study could be a starting point for further investigations about the relationship between radiological and surgical anatomy.

All authors conceptualized the study. P.T. wrote the manuscript and prepared the original draft. PG reviewed and edited the manuscript. F.M., L.G., C.D.C., and L.P. supervised the study. P.T., P.G., and L.G. confirm the authenticity of all the raw data. All authors have read and approved the final manuscript.

None declared.

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

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

The patients have provided written informed consent for the publication of any associated data and accompanying images.

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