The experience of financial burden for people with multimorbidity: A systematic review of qualitative research

Abstract Background Multimorbidity prevalence is increasing globally. People with multimorbidity have higher health care costs, which can create a financial burden. Objective To synthesize qualitative research exploring experience of financial burden for people with multimorbidity. Search strategy Six databases were searched in May 2019. A grey literature search and backward and forward citation checking were also conducted. Inclusion criteria Studies were included if they used a qualitative design, conducted primary data collection, included references to financial burden and had at least one community‐dwelling adult participant with two or more chronic conditions. Data extraction and synthesis Screening and critical appraisal were conducted by two reviewers independently. One reviewer extracted data from the results section; this was checked by a second reviewer. GRADE‐CERQual was used to summarize the certainty of the evidence. Data were analysed using thematic synthesis. Main results Forty‐six studies from six continents were included. Four themes were generated: the high costs people with multimorbidity experience, the coping strategies they use to manage these costs, and the negative effect of both these on their well‐being. Health insurance and government supports determine the manageability and level of costs experienced. Discussion Financial burden has a negative effect on people with multimorbidity. Continuity of care and an awareness of the impact of financial burden of multimorbidity amongst policymakers and health care providers may partially address the issue. Patient or public contribution Results were presented to a panel of people with multimorbidity to check whether the language and themes ‘resonated’ with their experiences.


| INTRODUC TI ON
Chronic disease, particularly multimorbidity (presence of two or more chronic diseases in a person), is one of the biggest challenges for health care systems globally. 1 The challenge of multimorbidity is primarily driven by the single-disease focus of health care systems, clinical guidelines and research. [2][3][4][5] The estimated prevalence of multimorbidity in the general population ranges from 13% to 72% depending on setting and age group studied 6 and has been increasing in recent decades. [7][8][9] Multimorbidity leads to greater health care utilization due to the extra health care needs associated with having additional conditions, but also the issues that arise as a result of the interactions between these conditions. 10 This is the primary cause of one of the central challenges of multimorbidity: the financial cost to health systems, society and the people who have multimorbidity. A systematic review of multimorbidity cost-of-illness studies concluded that multimorbidity was always associated with higher out-of-pocket (OOP) costs compared with 'non-multimorbidity'. 11 Multimorbidity has also been found to be associated with between five and ten times higher OOP costs for medications than no chronic conditions. 12 These high costs raise equity concerns, as multimorbidity disproportionately affects people from lower socioeconomic groups. 13 The World Bank reports that every country in the world imposes some form of OOP payments for health care on people 14 and these have been increasing. 15 People across the world also incur other costs when accessing health care (eg transport costs) and may experience indirect costs such as reduced income from employment due to treatment-related absenteeism. Multimorbidity is also a significant issue in low-and middle-income countries (LMICs), albeit prevalence is not as high. 16 A systematic review 17 concludes that financial catastrophe due to non-communicable diseases is evident across all continents and across all income strata. However, citizens of LMICs are more vulnerable to impoverishment due to OOP payments. 18 Along with impoverishment, the financial burden associated with multimorbidity can have other negative effects including reduced medication adherence due to inability to purchase medication 12 and reduced quality of life. 19 The financial burden associated with multimorbidity may also have an effect on health care utilization and contribute to the higher levels of mortality 20 and morbidity people with multimorbidity experience.
Many studies have examined costs associated with multimorbidity. 11 However, these primarily examine the cost of multimorbidity to the health system and not the cost to the individual. 11,21 Given that people's experiences are considered key in evaluating the quality of health care, 22,23 by synthesizing many qualitative studies, people with multimorbidity can be given a greater voice 24 and their experience of financial burden can be elucidated. The authors therefore aimed to synthesize qualitative research exploring experience of financial burden for people with multimorbidity.

| Study design
A systematic review of qualitative studies using thematic synthesis examining the experience of financial burden for people with multimorbidity was conducted, in order to provide a comprehensive picture of people's experiences to inform discourse and decision making. 25 A protocol detailing the methods 26

| Search strategy
The full search strategy is detailed in the protocol. A pre-planned search was conducted in six databases ( available, the corresponding author was contacted by email with one follow-up. If no reply was received within one week of followup, the study was excluded.

| Study selection
Studies were included if they used a qualitative design, conducted primary data collection, included first-or second-order references to financial burden and had at least one community-dwelling adult (≥ 18 years) participant with two or more chronic conditions (see Table 1). Studies exploring people's experience of 'chronic disease', where no specific condition was the focus of the study and many of the participants had one condition only, were included. First-or second-order references to financial burden were only included if it was clear that it was in relation to participants with multimorbidity.
Studies that focused on people with a single specific chronic condition were excluded.

| Screening
Search results were exported to EndNote X8, and duplicates were removed and then imported into Covidence. Firstly, one reviewer (JL) screened titles to remove entries clearly unrelated to the research question. Then, two reviewers (JL and LF) screened titles and abstracts independently, according to the inclusion/exclusion criteria (Table 1). Finally, full texts were reviewed independently by two reviewers (JL, LF). At both stages, disagreements were resolved through discussion. If agreement was not reached, then a third reviewer (BC or SS) decided on inclusion.

| Data extraction
One reviewer (JL) extracted study characteristics using a prespecified pro forma. 28 Extraction was cross-checked by a second reviewer (BC). Data for the thematic synthesis were extracted from the results sections only by one reviewer (JL) and crosschecked by a second reviewer (LF). Where studies presented views of participants other than people with multimorbidity, data were only extracted where it was clearly attributed to a participant with multimorbidity.

| Data analysis
Data were analysed using Thomas and Harden's method of thematic synthesis, 30 an inductive method used to draw inference based on common themes from studies with different designs and perspectives. 30 The following three-step process for thematic synthesis was conducted. First, one author read and re-read the included studies while conducting line-by-line coding. Secondly, these codes were grouped into related areas to form descriptive themes. Thirdly, these descriptive themes were iteratively examined and compared to refine the relationship between them and to generate themes that go beyond the descriptive themes to provide new insights related to the review question (analytical themes). NVivo version 12 was used for analysis.
This three-step process was carried out by one author (JL) and cross-checked by a second author (BC). Direct quotations from study participants are presented in italics to distinguish them from second-order data (author interpretations).

| Public and Patient Involvement (PPI)
In order to increase the credibility of the findings, the results were presented to a PPI panel with experience of living with multimorbidity. While acknowledging that they are often occurring in another context, this process offered a modified form of 'respondent validation' 31 in allowing the PPI contributors to check whether the language and themes 'resonated' with their experiences.

| Appraisal of studies
The critical appraisal skills programme (CASP) qualitative checklist 32 was used to assess the quality of included studies. Studies were independently evaluated by two reviewers (JL and BC). Differences were resolved through discussion. Studies were not excluded or weighted based on quality appraisal.

| Certainty of the evidence-CERQual assessment
The GRADE-CERQual approach 33 was used to summarize the authors' confidence in the reviews findings. This assessment was conducted by one reviewer (JL) and double-checked by a second reviewer (BC). Details of this process are in the protocol. 26

| Reflexivity
Details of the researchers personal worldviews and experiences are in the protocol. 26 The researchers reflected on their personal worldviews and experiences throughout the research process.

| Protocol deviations
Non-English studies were excluded. The CERQual assessment and data extraction were cross-checked by a second reviewer instead of being conducted in duplicate. A third reviewer did not oversee the analysis or the process of critical appraisal.

| Search results
In total, 22,580 citations were identified from searching the databases, and 2,146 identified from grey literature searches and forward and backward citation checking of included studies. After removing 9,955 duplicates, 14,771 records were screened, of which 1,900 were excluded based on title, and 12,511 were excluded based on title and abstract. Three hundred and sixty full texts were screened and 314 were excluded (Appendix B), leaving 46 studies included for qualitative synthesis (Figure 1).

| Quality appraisal
All included studies had a clear statement of the aim (see Figure 2 and Appendix C). While a qualitative methodology was appropriate in all included studies, it was unclear whether the qualitative design used was appropriate to address the research aims in 15 of the 46 studies. 38,40,42,43,48,50,55,57,63,64,69,71,73,76,79 This was primarily because the researcher had not provided a justification for the design.
In 28 of the 46 included studies, there was no consideration given to the relationship between researcher and participants and the potential bias that may arise in data collection, analysis and interpretation. 34

| Certainty of the Evidence
Using the GRADE-CERQual, 33 certainty of the evidence for individual findings ranged from low to high (Appendix D). There was consistency in most findings across countries and settings. Confidence in review findings were downgraded primarily due to methodological limitations and relevance. The main methodological limitation was in relation to inadequate exploration of reflexivity. The main issue with relevance was the high proportion of studies from the United States.

| Overview of results
Four descriptive themes related to the experience of financial burden were generated: (a) high costs, (b) access and negotiating health insurance and government supports, (c) coping strategies to manage costs and (d) reduced well-being. Table 2 reports participant quotations that are representative of these themes and their subthemes.
High costs were central to participants' descriptions of their experiences of financial burden. Whether the costs experienced by a participant were manageable and how high these costs were was determined by the participant's level of health insurance and government supports. When health care costs were unaffordable, participants had to access informal supports or make sacrifices. Being unable to afford health care and the associated sacrifices led to a negative impact on well-being for some participants.

| High costs
This theme related to the scale and detail of costs associated with multimorbidity and was discussed in 36 studies. The costs were reported either in terms of direct costs (costs directly related to care such as medicines, transport and health care appointments, discussed in 32 studies) or indirect costs (costs indirectly related to care such as loss of income, discussed in 14 studies).

| Health insurance and government supports
Health insurance (private and public) and government supports often determined whether the costs participants experienced were manageable and how high these costs were. Three subthemes were generated in which health insurance and government supports affected financial burden: insufficient coverage, safety net and complexity. Government supports also included social welfare payments.

| Insufficient coverage and insufficient care
For some participants, social welfare payments could not cover their health care costs along with their other basic needs. 40,48,53 Several participants talked about not being able to afford health insurance, 39,53 while others discussed making sacrifices (described in more detail below), including selling their home, because they felt health insurance was vital. 39 In some cases, participants felt that their lack of health insurance and/or government supports meant they had to wait for care 41 36,54 or their health insurance. 42 In one example, there were waiver programmes available that people did not know they could access. 36 In two studies, participants described being told that a service or medicine they were using was no longer covered by their health insurance. 51,65 In one case, there was a lack of clarity on how much a procedure was going to cost until it was carried out. 34 These issues led participants to seek out help from health care workers, 53 challenge the system by contacting the services responsible, 51 protest 41 or go to court to resolve issues. 72

| Coping strategies to manage costs
This theme related to coping strategies people with multimorbidity developed to manage high costs.

| Accessing informal supports
Thirteen studies mentioned help from family and friends as an informal support to manage the high costs associated with multimorbidity. 37 50 Costrelated non-adherence even occurred for prescription medicines with very small co-payments, due to the cumulative cost for many medicines. 40,55,69 One study described a person who, due to unaffordable costs, would only take medication when they were feeling unwell or to control symptoms. 65 High costs also led to some people not attending health care appointments. 36,37,40,49,52,54,[57][58][59]61,65,70,74 A US-based study reported that this was more likely to occur for black people. 68 Five studies reported that this was more likely to occur for people from deprived populations. 45,61,62,68,75 Authors also reported that black people 68  Participants also discussed losing their savings, 37,53 losing their home 39 and accruing high levels of debt in order to meet the high costs associated with multimorbidity. 53,72,75

| Reduced well-being
Fourteen studies reported that the costs associated with multimorbidity had a negative impact on well-being. 36,37,39,40,50,53,54,59,62,63,72,75,78,79 This manifested itself in many forms such as upset, 59 Not being able to work and the financial burden associated with this also had a negative impact on well-being, 36,79 particularly on people's self-esteem.
Financial burden left some feeling socially isolated because they could not afford social activities. 36,79 People also experienced shame and stigma because of their inability to pay for health care and the poverty they were experiencing. 36,39,53,79 Four studies 38,39,43,72 discussed people's experiences of relief at being spared financial hardship due to multimorbidity.

| Analytical themes
Analytical themes were developed based on the descriptive themes and the research questions specified in the protocol 26 : were being managed'. 38 Participants described having to travel to several different clinicians, return to the same health care facility several times due to lack of coordination of their care and pay extra for longer consultations due to multiple issues. All other areas of financial burden discussed above are directly related to the costs experienced by the person, and therefore, it is likely that multimorbidity exacerbates these areas of financial burden. For example, cost-related medication non-adherence is highlighted as a big issue Due to the complexity of accessing financial supports some people have to use different methods to navigate the health care system. and financial burden.

| Main findings
This systematic review of qualitative research identified 46 studies (45 datasets) from six continents. Financial burden was found to comprise the high direct and indirect costs associated with having multimorbidity, the coping strategies people have to use to manage these costs, and the effect of these costs and associated strategies on people's well being; government supports and health insurance often determined the manageability and levels of costs experienced.
These phenomena were found to exist across settings, including settings with universal health coverage (UHC). Using the GRADE-CERQual 33 approach, the certainty of the evidence ranged from low to high, but was moderate for most findings.

| In the context of other research
Many people with a chronic disease experience financial burden.
However, the results of this review highlight that multimorbidity increases costs and in turn exacerbates other areas of financial burden. The manner in which multimorbidity increases costs has been found across other studies: multimorbidity is associated with greater health care utilization, 80 higher rates of polypharmacy, 81 fragmented care 5,82 and an increased likelihood of leaving paid employment. 83 The results presented here provide insights into how these increased costs are experienced by people with multimorbidity.
Studies suggest that socio-economically deprived groups with multimorbidity are more vulnerable to financial burden. 84,85 This is exemplified in the findings which showed that deprived populations were more vulnerable to cost-related non-adherence. Given that socio-economically deprived groups are often found to be more likely to have multimorbidity, 13,86 interventions and resources may benefit socio-economically deprived groups most.
The findings reinforce that financial burden compromises the health of people with multimorbidity through non-adherence to medication and self-management practices and non-attendance at health care appointments, which could create a negative cycle. The components of this negative cycle have been documented in other research. [87][88][89][90][91] The results also show that financial burden compromises the health of people with multimorbidity through its impact on well-being, causing people to feel stressed, upset, worried and frustrated.
Despite the inclusion of countries with vastly different levels of health care access and government supports, most themes existed across countries. For example, insufficient coverage and insufficient care were evident across six continents and in countries such as Kenya 59 and England. 48 What may differ greatly between countries is the proportion of the population and sub-groups within it who are vulnerable to financial burden and the degree of impact financial burden may have on these populations. Levels of poverty are greater in LMICs, 92 and people in LMICs are more vulnerable to impoverishment due to OOP payments. 18 There were a disproportionate number of studies included from the United States which played a role in reducing the certainty of the evidence. One theme that was primarily evident in North America was complexities of health coverage. This is consistent with evidence that the United States has a complex health financing system, 93 though it may also be because there is a disproportionate amount of multimorbidity research from the United States. 2

| Implications
Despite the financial burden faced by people with multimorbidity, Patel and colleagues 94 suggest that clinicians are unlikely to raise the issue of financial burden with patients. This may be due to the wide range of issues faced by people with multimorbidity along with short consultation times. 95 Health care workers are often unaware of the issues people face with the treatments they are prescribed [96][97][98] and have been found to focus more on patients' biomedical information than their psychosocial information. 99 However, there is evidence that some clinicians, such as GPs, 94,100 are aware of the financial burden associated with multimorbidity.

| Strengths and limitations
The review protocol was published 26 and registered on PROSPERO.
The review was reported according to ENTREQ guidelines. 27 The study comprised a broad disciplinary team, which many multimorbidity studies are lacking. 21 The search strategy was broad. Thematic synthesis was an appropriate method of data analysis as it is used for studies with 'thin' data and analysis. 30 The use of PPI was novel and added to the credibility of the findings. Two reviewers for screening reduced bias. This review offers a counterbalance to the disproportionate amount of literature focussing on the cost of multimorbidity to the health system. 21 The studies identified were relatively recent, with 38 of the 46 included studies published between 2010 and 2019; therefore, the findings are likely applicable to current care.
The exclusion of non-English studies represents a bias. However, given that meaning may be lost in translation 108 and that there was breadth of countries and contexts covered by included studies, it was not a significant bias. The primary search was conducted over one year ago. However, given the extent of the search strategy and the large number of studies included from a variety of contexts, the authors concluded that the addition of new studies was unlikely to substantially change the findings. Despite the broad search strategy, some studies may not have been retrieved due to difficulties accessing qualitative literature. 109

| CON CLUS ION
The direct and indirect costs associated with multimorbidity are the fundamental components of financial burden for people with multimorbidity. However, this review highlights that financial burden is not simply the costs associated with multimorbidity, but also the coping strategies people use to manage costs and the negative effect both of these have on well-being; government supports and health insurance often determined the manageability and level of costs experienced.
Considering participants' many references to insufficient health coverage, UHC has the potential to reduce financial burden for people with multimorbidity. Greater consideration amongst policymakers and health care workers of all costs associated with accessing treatments can also mitigate financial burden. Finally, greater continuity of care can increase care coordination and reduce health care utilization, thus reducing financial burden for people with multimorbidity.

ACK N OWLED G EM ENTS
The authors would like to acknowledge and thank the public and patient representatives who provided constructive and informed feedback on the results of this study.

CO N FLI C T O F I NTE R E S T
The authors declare no conflicts of interest.

DATA AVA I L A B I L I T Y S TAT E M E N T
The data that support the findings of this study are available in the supplementary material of this article.