Keywords
Multimorbidity, costs, financial burden, qualitative systematic review, protocol
Multimorbidity, costs, financial burden, qualitative systematic review, protocol
Chronic disease has become one of the biggest challenges for healthcare systems globally1. This has brought into focus the phenomenon of multimorbidity, the presence of two or more chronic diseases in a patient2. Multimorbidity is of increasing concern due to the high disease burden and the related high rates of healthcare utilisation. The estimated prevalence of multimorbidity in the general population ranges from 13% to 72%2. These variations are largely accounted for by differences in settings and age groups across prevalence studies. The prevalence is likely increasing due to the ageing of the population globally3. Despite this, healthcare systems internationally are primarily single disease focused4. This single disease focus is reflected in clinical guidelines, which primarily treat diseases in isolation and rarely account for patients with multimorbidity. This creates a significant treatment burden4 which has several consequences for patients with multimorbidity, including a financial burden.
Financial burden refers to direct medical costs, direct non-medical costs and indirect-costs accruing to patients as a result of their multimorbidity. The financial burden of multimorbidity on patients is widespread and can be significant. A systematic review of cost-of-illness studies concluded that multimorbidity was always associated with higher out-of-pocket (OOP) expenditure than single or no chronic conditions5. Another systematic review found that a greater number of conditions present in a person was associated with higher OOP expenditure on medications6. This financial burden is of particular concern in terms of equity, as multimorbidity disproportionately affects patients from lower socioeconomic groups7.
Much of this economic-burden associated with multimorbidity arises from OOP expenditure or direct medical costs but it may also arise from direct non-medical costs including transportation to healthcare appointments and indirect costs including work absences. The economic-burden associated with multimorbidity can have negative effects including reduced medication adherence primarily due to inability to purchase required medication6, impoverishing spending (i.e., spending that pushes a household below an agreed poverty line)5, and reduced quality-of-life5.
Several qualitative studies have examined patients’ lived experience of multimorbidity8,9. Many of these studies have a brief focus on experience of financial burden. By synthesising these studies, a broader picture of this experience can be provided. It has been suggested that by synthesising many studies the patient is given a greater voice10. The authors therefore sought to synthesise qualitative research exploring experience of financial burden for patients with multimorbidity.
What are the experiences of patients with multimorbidity of financial burden?
How does financial burden affect interactions between patients with multimorbidity and the healthcare system?
How does financial burden impact on treatment burden for patients with multimorbidity?
There are recognised challenges in upholding the complexity and context of primary qualitative research when conducting a qualitative systematic review. However, patients’ views and experiences should inform decision making11 and these can be ascertained using qualitative methods11. By providing a systematic review and synthesis of this research, policy-makers can be more comprehensively informed11.
This review will be conducted and reported using the ENTREQ guidelines12. The review protocol is written in accordance with the PRISMA-P guidelines (reporting guidelines13).
The following databases will be searched: Lilacs, PubMed, CINAHL, EMBASE, PsycINFO, and Applied Social Sciences Index and Abstracts. Additionally, forward and backward citation checking of included studies will be conducted. Content experts will be contacted requesting information on any articles the content experts feel are relevant. For the grey-literature search a list of websites considered relevant by the research team were chosen (extended data13). Databases will be searched from inception using combinations of Mesh terms and key-words (extended data13).
Search results will be exported to Endnote X8 and Covidence, and duplicate entries removed. Initially, titles will be screened by a single reviewer (JL) to remove entries that are clearly unrelated to the research question. Then, two reviewers (JL, LF) will screen titles and abstracts independently; according to the inclusion criteria (Table 1). Any disagreements will be resolved through discussion. If this does not lead to agreement, then a third reviewer will decide on inclusion for full-text review. The same screening and conflict resolution processes will be carried out for full-text articles for inclusion in the analysis stage.
Only studies using a qualitative design, with primary data collection, referencing experiences of financial burden, and examining community-dwelling adults (≥ 18 years) with multimorbidity will be included (Table 1). Studies examining patients with non-specific chronic disease will be included if they include patients with multimorbidity and do not have a single condition focus. Qualitative design refers to studies which use a method of data collection and data analysis which are recognised qualitative methods14, for example interviews, focus groups, thematic analysis, and content analysis. Financial burden refers to the direct medical costs, direct non-medical costs and indirect costs experienced by patients. It is expected that the focus of studies will not exclusively be financial burden. Therefore, papers with broader focuses, such as the experience of multimorbidity, will be reviewed for inclusion. Also, many studies concerning financial burden and multimorbidity relate to polypharmacy, therefore studies concerning the experience of polypharmacy for patients with multimorbidity will be included if they meet the inclusion criteria. Studies without reference to financial burden will be excluded. Only data related to experience of financial burden for patients with multimorbidity will be included in the analysis. Mixed-methods studies with primary qualitative data collection will be included if they meet the inclusion criteria and where it is possible to extract the findings derived from the qualitative research. No language restrictions will apply.
Two reviewers will extract study characteristics independently using a proforma (see extended data13) under the following headings: setting, country, year of publication, methodology, participants (age, gender, socioeconomic status, conditions), sampling strategy, data-analysis technique, and definition of financial burden. Conflicts will be resolved by a third reviewer (BC).
Data (quotes, themes and author interpretations) will be extracted verbatim from the results section of included studies. This process will be conducted by a single reviewer (JL), and then cross-checked by a second reviewer (LF) to increase confirmability. Only data considered relevant to the research questions will be extracted. If information is unavailable from the full-text, then the corresponding author will be contacted for clarification. If there is no reply, then a follow-up email will be sent one week later and if no reply is received within one week of the second email then a decision will be made on inclusion based on information available.
Thematic-synthesis, as described by Thomas and Harden16, will be used. Thematic-synthesis is an inductive approach which is often used for studies with ‘thin’ data and analysis16. It is also used to draw inference based on common themes from studies with different designs and perspectives17. Thematic-synthesis consists of a three step process; step one consists of line-by-line coding of the data of included studies. The second step involves organisation or grouping these codes into related areas to construct ‘descriptive’ themes. In step three, the descriptive themes will be iteratively examined and compared to refine the relationship between them and generate analytical themes that is, themes that go beyond the descriptive themes to provide new insights related to the review question. Data will be coded using NVIVO version 12. Following multiple readings of the included papers data-analysis will be carried out by a single reviewer (JL) following the three steps outlined above. In order to increase confirmability of the analysis, all studies will be independently read by a second reviewer (LF) to crosscheck the coding structure and themes developed. This process will be overseen by a third reviewer (BC). In order to increase the credibility of the findings, an overview of the results will be brought for discussion to a panel of public and patient representatives with experience of multimorbidity.
The critical appraisal skills programme (CASP) checklist for qualitative research18 will be used to assess the methodological quality of all included studies. Two reviewers (JL and LF) will independently evaluate each study and any differences will be resolved through discussion. If this does not lead to agreement, then a third reviewer (BC) will adjudicate. Studies will not be excluded based on quality-appraisal. Quality-appraisal will be used as a means of discussing the quality of the included studies and to inform the GRADE CERQual (Confidence in the Evidence from Reviews of Qualitative research) assessment of confidence in the review findings19.
The review is intended to form part of a project which will inform how the specific needs of patients with multimorbidity are considered within clinical guidelines in Ireland. Therefore, the GRADE CERQual approach will be used to summarise our confidence in the evidence19. Four components contribute to an assessment of confidence in the evidence for an individual review finding: methodological limitations, relevance, coherence, and adequacy of data. Confidence in the evidence will be graded as high, moderate, low, or very low. This assessment will also be conducted in duplicate (JL and LF) and discussed amongst the research team.
Reflexivity. It is important to consider all findings in the context of research team members’ personal worldviews and experiences. Three authors have a background in social science; in psychology (JL, LF) and sociology and health services research (BC). One author has a background in general practice (SS) and is a leading expert in multimorbidity research, and one has a background in pharmacy and health economics (PH). The authors have operated within the Irish and other healthcare contexts. In relation to analysis, the lead researcher conducting the analysis (JL) does not have any chronic conditions. The authors will examine and discuss their preconceptions and beliefs surrounding the research questions, and consider the relevance of these preconceptions during each stage of analysis.
Dissemination of information. The review will be published in a peer-reviewed journal, reported using the ENTREQ guidelines12. The review will also be presented at a relevant conference and disseminated to policy-makers, patients, and the public.
Study status. At time of publication the study is ongoing, and title and abstract screening is underway.
The review will add to the knowledge base of how financial burden affects patients with multimorbidity as well as informing potential policy and practice interventions for patients with multimorbidity. This review also forms part of a project which, as a whole, will contribute to developing guidance of how the specific needs of patients with multimorbidity are considered within clinical guidelines in Ireland, and internationally. The review will inform the development of a national survey that will quantify economic burden for patients with multimorbidity in Ireland. Limitations include the potential paucity of data in included studies.
Open Science Framework: The experience of financial burden for patients with multimorbidity: A protocol for a systematic review of qualitative research: Extended data. https://doi.org/10.17605/OSF.IO/PN42R13
This project contains the following extended data:
Proforma.docx (a proforma with all headings under which study characteristics will be extracted)
Medline (OVID) Search strategy.docx (The mix of key words and mesh terms that will be used to search Medline and that will be transferred to other databases for searchers)
Grey literature search.docx (the list of websites that will be searched for grey literature using a variation of the Medline search strategy)
PRISMA-P checklist for ‘The experience of financial burden for patients with multimorbidity: A protocol for a systematic review of qualitative research’, https://doi.org/10.17605/OSF.IO/PN42R18
Data are available under the terms of the Creative Commons Zero “No rights reserved” data waiver (CC0 1.0 Public domain dedication).
Health Research Board Ireland [CDA-2018-003]
The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.
Is the rationale for, and objectives of, the study clearly described?
Yes
Is the study design appropriate for the research question?
Yes
Are sufficient details of the methods provided to allow replication by others?
Yes
Are the datasets clearly presented in a useable and accessible format?
Not applicable
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Research relevant to this review: Systematic review including metasynthesis, chronic and medically complex conditions in childhood.
Is the rationale for, and objectives of, the study clearly described?
Yes
Is the study design appropriate for the research question?
Yes
Are sufficient details of the methods provided to allow replication by others?
Yes
Are the datasets clearly presented in a useable and accessible format?
Not applicable
References
1. Eton DT, Ridgeway JL, Egginton JS, Tiedje K, et al.: Finalizing a measurement framework for the burden of treatment in complex patients with chronic conditions.Patient Relat Outcome Meas. 2015; 6: 117-26 PubMed Abstract | Publisher Full TextCompeting Interests: I have published in the areas of treatment burden and multimorbidity.
Reviewer Expertise: Treatment burden, multimorbidity, stroke.
Alongside their report, reviewers assign a status to the article:
Invited Reviewers | ||
---|---|---|
1 | 2 | |
Version 2 (revision) 26 Mar 20 |
read | |
Version 1 25 Jul 19 |
read | read |
Provide sufficient details of any financial or non-financial competing interests to enable users to assess whether your comments might lead a reasonable person to question your impartiality. Consider the following examples, but note that this is not an exhaustive list:
Sign up for content alerts and receive a weekly or monthly email with all newly published articles
Register with HRB Open Research
Already registered? Sign in
Submission to HRB Open Research is open to all HRB grantholders or people working on a HRB-funded/co-funded grant on or since 1 January 2017. Sign up for information about developments, publishing and publications from HRB Open Research.
We'll keep you updated on any major new updates to HRB Open Research
The email address should be the one you originally registered with F1000.
You registered with F1000 via Google, so we cannot reset your password.
To sign in, please click here.
If you still need help with your Google account password, please click here.
You registered with F1000 via Facebook, so we cannot reset your password.
To sign in, please click here.
If you still need help with your Facebook account password, please click here.
If your email address is registered with us, we will email you instructions to reset your password.
If you think you should have received this email but it has not arrived, please check your spam filters and/or contact for further assistance.
Comments on this article Comments (0)