Self‐management in older people living with cancer and multi‐morbidity: A systematic review and synthesis of qualitative studies

Many older people with cancer live with multimorbidity. Little is understood about the cumulative impact of old age, cancer and multimorbidity on self‐management. This qualitative systematic review and synthesis aimed to identify what influences self‐management from the perspective of older adults living with cancer and multimorbidity.


| INTRODUCTION
Cancer incidence is strongly correlated with age, with nearly 50% of individuals diagnosed with cancer aged over 70 years. 1 At any age, cancer can have a substantial impact on an individual's independence, as treatment side-effects can reduce the ability to maintain normal daily activities. Almost a quarter of those living with and beyond cancer (LWBC) report poor health or disability after primary cancer treatment. 2 Individuals can experience side-effects such as pain, breathlessness, and fatigue, as well as psychological problems including anxiety, depression and loss of confidence. 3 However older adults are at increased risk of the side-effects of some cancer-related treatments and common features of ageing may be aggravated by cancer treatment. 4 Older adults often lack the physiological reserves required to effectively recover from acute toxicities. In turn, this leads to ongoing problems related to quality of life (QoL) (a subjective concept, with a multidimensional perspective that encompasses physical, emotional, and social functioning 4 ). 5 In addition, older adults are more likely to have poorer literacy and numeracy skills, with less access to transportation, social support, or financial resources compared to younger adults LWBC. 6 Consequently, older adults may find it difficult to selfmanage symptoms, complex therapeutic routines, self-monitoring, self-assessment and interactions with healthcare providers and organizations. Self-management refers to "the care taken by individuals towards their own health and wellbeing: it includes the actions people take for themselves…to care for their long term conditions." 7 While cancer is a substantial health challenge, in older age it is often diagnosed in the context of other health needs that can complicate diagnosis, treatment, and management. 8,9 Over 75% of people with cancer report at least one other condition, and multimorbidity (defined here as the co-existence of two or more conditions) increases with age. 10 Older adults LWBC are more likely to have pre-existing conditions and to experience poorer physical functioning than younger people with cancer 4,9 and frequently report long-term support needs for management of complex health conditions after cancer treatment. 11 The number of people living with multimorbidity is rising with an ageing population. 12 Further conditions are also likely to develop after-and perhaps as a consequence of cancer and its treatment. These include diabetes, cardiovascular disease, neuropathy, or renal impairment. 9, 10 Leach et al 13 reported that older adults have an average of five long-term conditions, two of which develop after a cancer diagnosis. The onset of these conditions may be a consequence of ageing, behavioural/genetic risk factors, or due to late/ long-term effects of cancer treatments. Additional conditions may negatively impact cancer recovery, longevity, and reduce QoL. 4 Little is understood about how ageing and multimorbidity interact and influence outcomes in the context of self-management in cancer specifically. Clinical trials in oncology have often excluded older people or people living with frailty or multimorbidity. 9,10,14 Thus, clinicians have little evidence to help treat this age group 14 with a scarcity of evidence-based treatment guidelines and challenges in extrapolating study findings. 9 This hinders our ability to understand the long-term impact of cancer and its treatment, particularly in the case of those who are living with multiple other conditions. 4 These individuals may find it difficult to self-manage the disabling effects of symptoms, complex therapeutic routines, self-monitoring, self-assessment and interactions with healthcare providers and organizations. 15 A review by Kenzik 11 identified patterns of health care use in older adults after cancer. These included long-term hospitalizations, long-term care facility placement after cancer treatment, or home health care needs after cancer. However, the authors did not find any studies that addressed the potential impact of cancer on long-term care needs of the older adults. Kenzik 11 identified a focus on the ageing population as a high-priority gap in the evidence. When older people have needs that arise from other conditions, these must be co-managed alongside their cancer, potentially leading to increased work and burden for the patient and their families. For example, the risk of polypharmacy (ie, taking multiple medications) is higher for people living with and beyond cancer because this group are often prescribed a considerable number of medications, for both cancer management and for supportive care. 4 This increases the risk of drug interactions, morbidity and adverse events, especially for older adults who may already taking a number of medications for other conditions.
In order to understand the complex experience of older adults, we drew on Shippee's cumulative complexity model (CCM). This model sets out a mechanism by which complicating factors at individual patient level interact to affect self-management, utilization of healthcare services and health outcomes (see Figure 1 below). 16 An imbalance between the workload of demands and capacity to address these is likely to result in poor outcomes. 16 As burdens accumulate and increase in complexity, patients may be less able to cope with demands placed on them. Self-management requires patients to balance the workload of everyday life with the workload of managing their own health, mobilize and coordinate resources, and manage any limitations to their own capacity to do this work. 16 Treatment and illness burdens compound the picture, with negative outcomes leading to further imbalance, resulting in complexity accumulating over time. 16 This synthesis analyses findings from multiple qualitative studies to inform understanding of how older adults manage complexity associated with multiple health conditions in the context of ageing and LWBC. The review question was: "What do older adults living with and beyond cancer and multimorbidity report as influencing self-management?" In line with the CCM, we focus here on patient capacity and burden of treatment. These constructs informed the search strategy and defined dimensions of interest for analysis.

| METHODS
Systematic review procedures were used for study selection and data extraction, together with a comparative thematic approach to analysis and synthesis. 17 Our focus on identifying studies that yielded qualitative data reflected a primary aim to review perspectives and experiences of living with old age, cancer and multimorbidity. In contrast to systematic reviews, qualitative syntheses of this kind aim to give deeper conceptual insight through the synthesis of a group of related studies, using induction and interpretation to mirror the qualitative methods of the individual studies included. 17 This review was written using guidelines from the Enhancing transparency in reporting the synthesis of qualitative research (ENTREQ) tool. 18

| SEARCH STRATEGY
A dearth of MeSH terms for multimorbidity necessitated an iterative approach to developing a search strategy. 19 We drew on methods outlined by Gallacher et al for conducting a qualitative systematic review of a novel construct. 20 We initially carried out a series of scoping searches to identify key articles that fitted our proposed inclusion criteria. This process included citation tracking (forward searching of studies that have cited the paper), reference tracking (backwards search of studies referenced in articles) and "berry picking" of "clusters" of similar papers often found together. We also used the "related articles" function in databases. We drew on strategies used in existing similar reviews to develop as comprehensive a search as possible. 19

| DATA COLLECTION AND ANALYSIS
Abstract and title screening of all papers was undertaken independently by TC and AC based on pre-specified inclusion criteria (Table 1).
Differences in judgement were resolved by JB. Full-text articles were obtained and assessed by TC and AC.
A data extraction sheet informed by the CCM was designed a priori. Extraction of data was carried out independently by AC and TC, with a 5%-10% cross-check to ensure consistency. The results/findings sections of each paper were extracted, to reflect participant views (not author's interpretations), and treated as formal data. We extracted data related to those participants who were identified as being aged 65 or older (or were considered "older adults" by the authors of the included study as per our inclusion criteria in Table 1), F I G U R E 1 A patient-centred model of patient complexity (from Shippee et al 16 ): The model emphasizes a central, individual-level mechanism whereby complicating factors influence care and outcomes: the balance between serviceusers workload of demands and patient capacity to address demands. Workload incorporates the demands on the person's time and energy, encompassing demands of treatment, self-care, and everyday life. Capacity relates to one's ability to manage work (eg, functional morbidity, financial/ social resources, literacy). Workloadcapacity imbalances are the mechanism driving patient complexity. Treatment and illness burdens serve as feedback loops, linking negative outcomes to additional imbalances, so that complexity can accumulate over time and who were identified as having another condition alongside their cancer diagnosis. When papers focused on the experience of living with specific comorbidities or multimorbidity, data were extracted related to those participants identified as having had cancer as one of their conditions. Papers that included data from family or other unpaid caregivers were also extracted for analysis if they referred to the lived experiences of older adults with multimorbidity. Results relating to health care professionals' (HCPs) views or experiences were not included.

| Quality assessment/Appraisal
Included articles were assessed using the Critical Appraisal Skills Programme (CASP) tool, 25 to consider the quality parameters of qualitative work. We did not exclude articles on the grounds of quality alone but relative quality of included studies was considered during analysis.
Quality assessment was undertaken by TC and AC during the data extraction process. Differences of opinion were discussed and reviewed by JB. See Appendix B in Data S1 for more information. Papers that reported on the experience of older adults with multimorbidity and any cancer type Focus on self-management after the completion of treatment for cancer. In papers that did not distinguish between those coming to end of treatment and those post-treatment, only included papers that indicated that at least some of the participants have completed primary treatment of cancer and are currently self-managing their illness in the community.
Did not include studies that focused solely on treatment-decision making, palliative care, or ongoing receipt of in-hospital cancer treatment.
Articles included the perspectives of older adult patients (aged ≥65 y 3 ) and informal caregivers of patients aged ≥65 y If sample included individuals both under and over 65: included those papers where it was possible to extract data on experiences of those over 65 Papers that referred to "older" adults or similar without providing an age range were considered for inclusion An informal caregiver was defined as "anyone who cares, unpaid, for a friend or family member who cannot cope without their support." 21 Data only extracted for analysis if it related directly to the lived experiences of older adults with multimorbidity.
Coexistence of multiple chronic diseases and medical conditions in the same individual (two or more conditions) 19 Definition of chronic disease: "health problems that require ongoing management over a period of years or decades" 22 Articles focusing on areas denoted in the dimensions of interest based on Cumulative complexity Model Patient workload: All tasks and responsibilities that people engage in on a day-to-day basis. Includes everyday life demands plus the responsibilities of patient-hood, including job/family, self-care, clinical appointments, and other priorities. 23 Patient capacity: Abilities, resources, or willingness to cope with demands. 16 Incorporates affective, cognitive, informational, material, physical and relational resources that can be mobilized by individuals, 16 for example: physical and mental functioning, socioeconomic resources, social support, literacy, attitudes/beliefs and self-efficacy. 23 Workload and capacity balance: Balance between the patient's workload of demands and patient capacity. 24 Focus on the imbalance between the two-workload that exceeds capacity Outcomes of imbalance: for example, poor access, non-adherence, low QoL, or other problems; experience of care as burdensome, fragmented, or low quality; poor engagement and health outcomes etc. 23 These constructs are outlined in the cumulative complexity model). 16 The model emphasizes a patient-level mechanism whereby complicating factors that impact the balance between patient workload of demands and patient capacity to address demands. These factors contribute to treatment and illness burdens which may lead to negative outcomes and lead to complexity accumulating over time). 16  we remained open to other interpretations and lines of enquiry, constantly testing the extent to which the CCM helped to interpret, characterise and explain the data. The unit of analysis was the theme rather than the prevalence or frequency of statements. Themes were discussed and iterated in workshops until consensus was reached. We used Microsoft Excel to manage records and data throughout the review.

| RESULTS
Thirty papers reporting 28 individual studies were included in the final analysis (see Table S1 for detail on included studies and Table S2 for Participant characteristics). Figure 2 depicts the PRISMA flow diagram of studies identified and excluded at each stage of the review.
The findings below illustrate how old age and multimorbidity can accumulate to complicate self-management and threaten health and well-being, adding burden and diminishing capacity. Five themes are presented and Appendix C in Data S1 provides a summary table of which studies contributed to which theme. Quotes are presented with as much detail about the participant as possible, based on the information provided in the original text from which it was taken. While space to present illustrative quotes s limited in this paper, many more supporting quotes can be accessed in Appendix D in Data S1.

| Burden and capacity for health-related work
Existing health-related work complicated engagement with self- There was a sense that being older contributed to social isolation, not simply due to cancer or illness. Many lacked a supportive network outside of their families that could potentially offer support. Participants described losses they were experiencing in their supportive networks, as many of their friends and close family were also falling ill and dying. Nonetheless, some noted that they had developed a new social network as a result of their illness that helped to buffer against loneliness and isolation. Others expressed a desire to engage with more support within the community but were unaware of how to access this.

| Missed potential for HCPs to reduce burden and enhance capacity
Many expressed a desire for appropriate and well-timed information from an approachable and trusted healthcare provider, and an opportunity to ask questions and raise concerns. It was evident they wanted to be treated holistically as individuals and not to be seen uniformly as to patient workload, as they felt obliged to act in a manner that would avoid perpetuation of stereotypes relating to older people.
He said to me, "Don't worry about that. A lot of people your age, something happens like this, and they continue with a cane forever. Just accept it. You know, you're not as young as you used to be." I could kick him right in the pants because that's one of his things: "You know, you're not as young as you used to be." And I told him once, "You know, if I believed that, I'd have been dead 10 years ago" (Participant quote: participant in their 80's 8 ).

| DISCUSSION
Challenges encountered because of old age and multimorbidity may accumulate to complicate self-management, threatening health and well-being, increasing burden and diminishing capacity. To manage, older adults prioritized self-management practices that aligned with their own beliefs and that they saw as most likely to result in continued independent living. Conditions with the greatest impact on independent living assumed the greatest importance, and so, for many, previous experiences of cancer assumed a low priority. Self-management practices seen as likely to interfere with QoL were deprioritized unless viewed as necessary to maintain independence. When burden outweighed capacity, people were reluctant to ask for help from their social network. The contribution of formal healthcare services to supporting self-management was viewed as relatively peripheral.
Previous research has also demonstrated that people with multiple conditions often prioritize a "main" long-term condition. 56 Our work builds on these findings by specifically focusing on the experience of cancer and concurrent ageing alongside multiple conditions.
The older adults in the included studies often did not prioritize cancer and did not perceive the condition as having an impact compared to health conditions that interrupted daily life. Interestingly, we noted that some respondents expressed skepticism about the relationship between cancer and health-related behaviours and in turn, the efficacy of recommended self-management strategies. In particular, the relatively invisible impact of self-management strategies recommended after cancer was contrasted with the immediate and evident impact of self-management of co-existing conditions such as diabetes or arthritis. Such views may conflict with policy and practice agendas that describe cancer as a "long term condition." Many perceived cancer as something that had happened it the past that did not have an ongoing impact. Cancer appeared to only be prioritized when it resulted in highly visible long-lasting side-effects. However, it was often difficult for older adults living with multimorbidity after cancer to ascertain the precise source of symptoms experienced. Previous research has indicated that some individuals prioritize conditions based on the immediate impact of the condition on functional health, whereas others focus on the significance of or the future risk associated with the condition. 56 In our study, cancer was regularly viewed as a potential future risk, even if it was not an interruption in everyday life. Even among those who viewed cancer as something that had been treated and was in the past, conversations about recurrence still prevailed. People frequently described cancer in relation to mortality, in a way that they did not when they described other long-term conditions. Cancer was conceptualized as a possibly acute or intermittent (albeit high-risk) condition, rather than an ongoing long-term condition.
Complex symptomology made it difficult for individuals to establish the source of, and make sense of, their symptoms. In particular, our focus older adults living with cancer and multimorbidity has demonstrated how the particular context of ageing can influence attitudes, beliefs, social support and coping strategies employed. The importance of lay beliefs about symptoms is useful to consider in the context of existing literature on those LWBC and co-existing multiple conditions. Such individuals are significantly more likely to report poor health outcomes than those with no history of cancer or a chronic condition. 57 Those with multiple conditions report significantly worse QoL than those with only one condition, which in turn is significantly worse than the healthy population. 57 Therefore, cancer does seem to add a degree of complexity, even if this complexity is not always recognized or articulated by the older person. However, we also identified the psychological "work" that individuals undertake to help them to understand, make sense of, and prioritize their conditions in order to enable more effective self-management.

Individuals' beliefs and previous experiences appeared to influence
how they thought about their various conditions. Psychological factors shaped individuals' readiness to perform patient-related work, and drive patient's overall response to self-management and cumulative complexity. Beliefs about (and attitudes towards) complexity appeared to be key drivers of health behaviours, influencing support and subsequent outcomes. This subjective response often appeared to play a more significant role than the influence of objective patient workload and capacity. In some cases, the beliefs people had about illness prior to a diagnosis of cancer may lead to feelings of acceptance and resilience that reduce the perceived burden of illness and treatment. This finding is in keeping with previous literature relating to social cognitive approaches to behaviour (eg, the work of Bandura 58 ).
For example, the common-sense model of self-regulation proposes that individuals form lay representations of illnesses that guide coping procedures to manage illness. 59

| Study limitations
As previously noted by Cavers et al, 10 identifying qualitative data on this precise topic was challenging as age and multimorbidity were often "hidden" in articles with a different prevailing focus. However, our stringent inclusion criteria developed apriori enabled us to select appropriate studies for inclusion. Further, while the included studies varied widely in their focus, our systematic analysis of this set of rich, qualitative studies has served to identify important themes that transcend individual study differences. The dataset was largely focused on the experiences of individuals with particular characteristics (ie, breast cancer, Caucasian, and female), and more research is needed to better understand the experiences of specific subgroups (specifically socioeconomically disadvantaged groups, those with low health literacy, and ethnic minorities), and crucially to those who may have more severe symptoms as a consequence of their cancer or another condition. The type of cancer that an older person is diagnosed with is likely to affect its impact on other health conditions. Those with breast and prostate cancer report lower comorbidity burden than those with other cancers that may lead to more disabling side effects, including leukaemia, colorectal, oral, and bladder cancer. 14 Recruitment of more heterogeneous samples in future studies of this nature is recommended, including a wider range of cancer types and treatment stages.
Synthesizing qualitative research can add to the findings of individual studies, however we are reliant on the content presented with study reports as opposed to the raw data from the individual studies.
Some have argued that original qualitative studies are interpretive and are drawn from data that are not intended to be generalisable nor without bias. 60 Conversely, some have suggested that qualitative synthesis should seek to achieve transparency rather than reproducibility. 61 To enhance transparency, we have provided detailed descriptions of the inclusion criteria, search strategies and details of included studies. Further, we have provided quotations from the raw data so readers can make their own judgements about the relevance and utility of the data (Appendix C in Data S1).

| Clinical implications
Our findings reinforce the view that post-treatment cancer care is often disjointed, with older patients facing challenges in liaising with healthcare professionals. Over-stretched healthcare services often struggle to manage care for complex patients and there is frequently a gap in healthcare provider expertise, patient-centred care and interprofessional collaboration. Patients are at risk of problems associated with polypharmacy, such as poor medication adherence and adverse drug-related effects. These individuals are often very conscious of clinicians' workload, with some reporting concerns about taking up too much time. Short clinic appointments required older adults to prioritize issues and individuals did not always feel comfortable raising concerns about seemingly vague or unexplained symptoms. This is in keeping with existing literature indicating that older people may be more likely to defer to authority and may not feel that they are able to gain information they need or ask questions. 62 Persistent symptoms associated with cancer may not be shared with health professionals. Further, the extent of the work (in particular added workload) that individuals do to manage their health after cancer might also remain unnoticed by health professionals, but perhaps also by the patients themselves. Without adequate support from (and access to) health care professionals, lay understandings may predominate, leading to misattribution of symptoms and missed opportunities to promote wellness. Our findings highlight a need to develop more collaborative approaches to supporting individuals that address older patients' priorities and needs after cancer. Truly person-centred approaches challenge practitioners to develop skills to communicate effectively, coordinate care, and be able to balance competing priorities in order to manage complicated consultations in a limited timeframe. 63 There is a need to move beyond a focus on a single chronic diseases and instead, move towards a more comprehensive approach that focuses on the cumulative impact of a number of conditions on daily activities and quality of life. 64,65