Conceptual framework for living with and beyond cancer: A systematic review and narrative synthesis

Abstract Objective The concept of living with and beyond cancer is now emerging in policy and literature. Rather than viewing this notion simply as a linear timeline, developing an agreed understanding of the lived experience of people affected by cancer will aid the development of person‐centred models of care. Methods A systematic review was conducted. The review question was “What does the term ‘living with and beyond cancer’ mean to people affected by cancer?” The protocol for the review was preregistered in the PROSPERO database (PROSPERO CRD42017059860). All included studies were qualitative, so narrative synthesis was used to integrate descriptions and definitions of living with and beyond cancer into an empirically based conceptual framework. Results Out of 2345 papers that were identified and 180 that were reviewed, a total of 73 papers were included. The synthesis yielded three interlinked themes: Adversity (realising cancer), Restoration (readjusting life with cancer), and Compatibility (reconciling cancer), resulting in the ARC framework. Conclusions Three themes describe the experience of living with and beyond cancer: adversity, restoration, and compatibility. The ARC framework provides an empirically informed grounding for future research and practice in supportive cancer care for this population.

with the effects of primary cancer treatment. 3 On the other hand, people affected by cancer can lean towards a different meaning, whereby living with and beyond cancer begins at diagnosis. 4 The need for clarity and an agreed understanding has therefore been identified to better understand the experiences and needs of those who are undergoing or have completed primary cancer treatment. This understanding can be used to develop person-centred models of care and improve the long-term quality of life for people who live with and beyond cancer. 5 A conceptual framework addresses this need by providing a synthesised understanding of living with and beyond cancer and to date, no systematic review of evidence on conceptualising living with and beyond (all types of) cancer (LWBC) has been undertaken. The aim of this study was to identify and synthesise disparate accounts of LWBC into a thorough, empirically based conceptual framework, with a view to providing an empirical basis for future research and practice in cancer care. 6

| METHODS
The research aimed to draw together literature to explore the meaning of LWBC. The protocol for the review was preregistered in the PROS-PERO database (PROSPERO CRD42017059860).

| Eligibility criteria
The review sought to identify conceptual and empirical papers that explicitly developed a conceptualisation of living with and beyond any form of cancer. A conceptualisation of LWBC was defined as a theory, model, or framework, which emerged from an analysis of primary data on at least three participants. 7 Debates on sample size and research quality continue; Pollio, Henley, and Thompson recommend, "although not a formal methodological rule, the situational diversity necessary for identifying thematic patterns is often provided by three to five interview transcripts." 8(p51) The aim of this study was to reflect the reality of participant accounts so a predefined understanding of LWBC was not used.
Studies were included if they 1. contained a conceptualisation of LWBC from which a succinct summary could be extracted; 2. presented an original model or framework (based on primary research) of LWBC; 3. presented primary research involving quantitative or qualitative data based on at least three participants; 4. were available in printed or downloadable form; and 5. were available in English.
Exclusion criteria were as follows: 1. studies focusing solely on living with the consequences of treatment, secondary symptoms, or dying; 2. studies focusing on health-related quality of life that used a predefined definition; and 3. studies defining clinical remission criteria or recovery from cancer.

| Search strategy and data sources
Three search strategies were used to identify relevant papers: electronic database searching, hand searching, and web-based searching. The search strategy was designed in OVID and modified for EBSCOhost and Elsevier and is shown in Table 1.

| Electronic database searching
Initial scoping searches were completed using three databases (PsycINFO, Medline, and CINAHL) to narrow the key words and to test medical subject heading (MeSH) terms. Due to the specificity of the search (ie, patient experience and cancer as a disease), medical subject headings were not used, and search terms were refined and modified to optimise the balance between specificity and sensitivity.
Limits were also placed on the protocol to ensure feasibility. For example, studies focusing on living with secondary symptoms or living with dying were not included.

| Hand searching
The tables of contents of journals which publish key articles (British Journal of Cancer, Psycho-Oncology, and Cancer Nursing) were hand searched (from 2000 to May 2017). These journals were chosen because they were identified (eg, in database search) as having published research specific to LWBC. While secondary research was excluded from the review, existing systematic reviews 9-16 and literature reviews 17 of living with and beyond cancer were also hand searched. 2 ("life" OR "live" OR "lives" OR "living") adj ("with" OR "beyond" OR "after") Living With and Beyond 3 "theor$" OR "framework" OR "model" OR "dimension" OR "paradigm" OR "concept$"

| Data extraction
Duplicates were removed in Endnote, Version 7. 18 Titles identified in the electronic search were screened, to identify those with possible relevance. Abstracts from relevant publications were reviewed, and where they appeared to meet the inclusion criteria, the full publication was obtained and assessed for eligibility. Of the 2280 abstracts identified in the database search, a random 15% (n = 342) were independently rated by authors C.L. and C.B. for eligibility.
One rater (C.L.) assessed the eligibility criteria for all 180 retrieved papers, with a random subsample of 85 papers independently rated for reliability by a second rater (C.B.). Acceptable concordance was predefined as agreement on at least 90% of ratings. A concordance of 92% was achieved. Reasons for exclusion were recorded, and disagreements were resolved through discussion or by a third rater (S. A.). For each included paper, the following data were extracted and tabulated: methodological approach, participant information and inclusion criteria, study location, and summary of main study findings.

| Quality assessment
All included studies were qualitative, so quality was assessed using an established framework for assessing qualitative research evidence. 19 The quality assessment covers the various stages and processes within qualitative enquiry, and the contribution, defensibility, rigour, and credibility of the study. One rater (C.L.) assessed the quality of all included studies, with a random 20% subsample of all papers (n = 15) independently rated by a second rater (C.B.). Consensus between raters was required, with differences in opinion on two of the 15 papers resolved through discussion. Spencer et al 19 clarify that the quality framework is to be used as a heuristic guideline and so in order to make judgements about the overall quality of papers a point score was calculated using the quality framework. 19 Each of the 18 items were weighted equally and rated "yes" (allocated 1 point) or "no" (allocated 0 points), giving a maximum quality rating of 18. The studies were divided into three groups; high quality was defined as a score of 13 or more, medium-quality papers scoring 7 to 12, and low-quality papers scoring 6 or less.
Quality assessment was not used to exclude papers given the debate on whether quality checklists rate the quality of the study or the quality of reporting. 20 Instead, quality rating was used for sensitivity testing. For example, similarities and differences in results were explored across high-quality studies as well as across all studies (high-, medium-, and low-quality papers). ings to develop a coding frame. Equal attention was paid to each data extract to identify initial codes, and codes were organised into one or several broader interpretive themes to fully capture their meaning. 22 Thematic maps, that are a visual representation of the themes, were used to organise the themes by clustering all codes according to connections in the data and by considering the patterns and relationships between themes. 22 Additional codes, refinements to the specifics of themes, and thematic patterns continued until theoretical saturation was achieved. Theoretical saturation occurred when the emergent themes had been fully explored and new data was easily accommodated within them. 22 Themes needed to be present in at least two studies to be included in the synthesis, and the themes were confirmed as being representative of the literature by a second analyst (L.M.).

| Stage 2: exploring relationships between studies
Vote counting was conducted to identify similarities and differences between each study, including a subgroup analysis by country, cancer type, and stage of illness. Thematic vote counting was also conducted using codes developed in the thematic analysis for this cross case comparison. 21 2.5.3 | Stage 3: assessing robustness of the synthesis They were asked to comment on the general language and use of an acronym to describe the framework, the positioning of concepts within different hierarchical levels of the conceptual framework, to identify any important areas of LWBC which they felt had been omitted and to make any general observations. The preliminary conceptual framework was modified in response to these comments, to produce the final conceptual framework.

| Ethics approval
The systematic review was conducted as part of a larger study, funded by Macmillan Cancer Support and hosted by Imperial College Healthcare NHS Trust. Ethical approval was obtained from the West

Midlands-Black Country Research Ethics Committee and the Health
Research Authority (REC reference 17/WM/0127).

| RESULTS
A total of 73 studies focusing on the personal perspective of LWBC were identified for inclusion in the review. The flow diagram for the 73 included papers is shown in Figure 1 and Data S1 lists those papers that were included.

| Quality assessment
The mean quality rating score for the 73 qualitative studies was 10.9 (range 3-17). A quality rating score of 13 or above, indicating high quality, was obtained by 25 (34%) papers. A quality rating score of 7 to 12, indicating moderate quality, was obtained by 42 (58%) papers, and a quality rating score of 0 to 6, indicating low quality, was obtained by six (8%) papers. The quality ratings of each included study are reported in the data extraction table (Data S3).

| Stage 1: developing a preliminary synthesis
Living with and beyond cancer is an experience that disrupts implicit assumptions about life and forces people to reconstruct their perspectives on self and future. [23][24][25][26][27][28][29][30][31][32][33][34][35][36][37][38] The disruption of living with and beyond cancer has been associated with both "relinquishing control" 39 and "taking charge," 40 and while living with and beyond cancer is associated with loss and altered life, [41][42][43][44][45][46][47][48] included studies also report positive changed attitudes and views of life after a cancer diagnosis. [49][50][51][52][53][54][55][56][57] The Included studies further highlight the adversity experienced because of healthcare. Some studies describe a "silent" health care system that provides limited time with healthcare professionals. 45,63 Others highlight the feeling of struggling and a loss of control due to the lack of participants' involvement in treatment planning:   Kucukkaya found an increased self-awareness, acceptance of old and renewed personality and increased appreciation of personal worth. 55 Offering peer support and a willingness to help others is identified as another support mechanism in the search for compatibil-

| Stage 2: exploring the relationships between studies
All 73 studies were included in the vote-counting process. For each dominant theme, papers were characterised using subthemes developed from the synthesis. Data S6 shows the vote counting for the subthemes of each of the three core themes. The "Adversity" subthemes present in the most studies were "life-changing impact of diagnosis" (58 studies) and "impact of treatment" (48 studies). The "Restoration" subthemes most frequently identified were "importance of social support" (30 studies) and "lifestyle changes" (28 studies). The most frequent "Compatibility" subthemes were "benefit finding" (15 studies), "offering peer support and willingness to help others," and "broader perspectives of well-being" (both 11 studies each).
Overall, included studies of personal experiences of living with and beyond cancer made reference to Adversity, Restoration, and/or Compatibility. All three ARC themes were identified in 19 of the 73 studies (26%), with the strongest mapping for "Adversity" (94.5%) and the weakest mapping for "Compatibility" (34%). Contextual aspects such as country, study setting, participant (eg, age), or type of cancer did not produce any apparent differences within the ARC themes. High- ness. 77 Charmaz, a medical sociologist and ethnographer, also presents the concept of "loss of self" as a central aspect of the experience of illness, beyond physical suffering. This refers to losing valued aspects of one's identity, due to a cascade of physical limitations and social changes. 78 This account is mirrored in the "Adversity" and "Restoration" themes, which describe efforts to manage and contain the impact of diagnosis, symptoms, and treatment on personal identity, with the responses of one's social and health care context being a significant influence. Equally, Brennan proposes a clinically useful conceptual model of psychological adjustment in cancer, describing a continuous and iterative process of adapting one's mental models (of self and future etc) when cancer experience disconfirms one's previous implicit versions. 79 The descriptions of a range of experiences within the "Adversity" theme align closely with this model. For example, even as people report reactions that are disparate on the surface (eg, cancer as "a massive shock" yet also cancer "not taking precedence"), they both derive from the same underlying process and thus can sit coherently within the same theme. In addition, the "Compatibility" theme relates to how people

| Clinical and research implications
The ARC framework challenges more traditional cancer models because it is built from studies of personal experience. While a chronic disease model of care 82 is widely adopted in the management of common chronic illness such as diabetes, depression, and heart failure (http://www.improvingchroniccare.org), the ARC framework questions the notion of chronicity associated with living with and beyond cancer and is a useful conceptual framework for translating the LWBC experience into shaping supportive cancer care.
Like patient pathways that cross organisational boundaries, the ARC framework can also be used across all levels of the health system and in primary and secondary care. 83 The ARC framework will is experienced and reexperienced and as the biopsychosocial impact of the illness unfolds, "patient education" needs to be delivered at multiple touch-points rather than just clinical starting points. Also, the ARC framework can express significant aspects of cancer experience without resorting to unduly medicalising or reductive language.
It can therefore empower patient representatives and advocates with a tool for promoting patient experience and a structure for its expression, thus increasing its impact. This may be especially relevant with regard to patient-centred care, which is often a service priority but can be clouded by complexities regarding specific clinical protocols. 84 Alongside, the ARC framework provides a foundation for structuring local guidelines and future policy, benchmarking clinical practice (eg, a basis for developing an accreditation process for services), and supporting staff development within existing practice competencies. 85 In relation to research, the ARC framework can provide a core foundational structure as a starting point for investigation in novel and under-researched areas, as well as challenging overly simple research questions. For instance, "Restoration" includes a significant social support dimension, and thus dyadic-or familywide rehabilitation may be more pertinent than more narrowly individual self-management, though much more complex to research. 86 Finally, at the level of clinical practice, the ARC framework can provide healthcare staff an evidence-based tool for interpreting patients' narratives and guiding them appropriately. For instance, "Compatibility" highlights how cancer experience is often deeply transformative, and thus clinicians who hear a patient hoping that "after treatment everything will settle down and be the same again" can usefully reframe LWBC as "a new normal" that often involves both welcome and unwelcome changes. The ARC framework is informing clinical training in primary care Improving Access to Psychological Therapies services and the secondary care Clinical Nurse Specialist workforce. An example of ARC use in direct clinical practice is as a conversation guide for clinicians and patients completing the recovery package. 87 For example, people are supported to identify interventions and assistance that would enable transition towards the readjustment and compatibility themes, as well as how they might be supported to manage any adversity, as part of their holistic needs assessment. Secondly, by adopting broad and inclusive criteria on study context (eg, service setting and cancer pathway), while checking (using narrative synthesis methodology) that these do not unduly influence the results, this study allows the emergence of a broader account of patient experience. Thus, this framework is arguably more personcentred and less subject to a priori influences and preconceptions of clinical parameters, policy priorities, or service realities. As a result, it can serve better as a solid basis for development in novel or underdeveloped areas (eg, a novel treatment or the experience of people with pre-existing psychosocial vulnerabilities). It can also constructively challenge more established areas (eg, cancer rehabilitation) to reflect on whether the practices and solutions that have developed still align well with core, shared themes of personal experience.

| Study strengths and limitations
Conversely, a potential limitation is that narrative synthesis is a secondary analysis of data based on existing interpretations by the authors of the original papers. As we necessarily accrue inferences and interpretations with every level of abstraction, there is potential for subtle or divergent accounts to be overlooked, reducing the richness of the account or inadvertently silencing some experiences. We safeguard against this through evaluating primary study quality, cross validating with clinicians and patients, and ensuring rigour throughout the research process. Nevertheless, we recognise that this account should be viewed as a heuristic framework, rather than as definitive and nomothetic. Similarly, it is important to note that while individual real-world experiences can be accommodated by the ARC framework, the synthesis of existing studies was at group-level and individual accounts go beyond the scope of this study.
A key challenge for health services is the lack of clarity around what constitutes living with and beyond cancer. The synthesis contributes to the understanding of living with and beyond cancer, and the emerging conceptual framework can be used to support clinical practice by identifying and responding to the needs of the personal experience of living with and beyond cancer.