Effectiveness, cost effectiveness, acceptability and implementation barriers/facilitators of chronic kidney disease management programs and models of care for Aboriginal and Torres Strait Islander Australians: a mixed methods systematic review protocol

Review objectives and questions The objective of this mixed methods review is to synthesize quantitative, economic and qualitative evidence on chronic kidney disease (CKD) management programs and models delivered to Aboriginal and Torres Strait Islander Australians. Studies with Indigenous participants from New Zealand and Canada will also be considered because similar persistent patterns of health inequities have arisen in these countries as a result of a shared colonial history, despite vast differences in timing and location.1,2 Also, there are geographic and demographic similarities, such as remoteness from health services and poor engagement due to differing language, culture and concepts of health and illness from the dominant culture. These socio-demographic circumstances are associated with higher burdens of chronic disease and poorer health outcomes.3,4 The intention of this systematic review is to inform CKD program design, practice and service delivery to Aboriginal and Torres Strait Islander populations in Australia. The questions to be addressed in the review are: What is the effectiveness of programs/models in relation to outcomes, including, though not limited to, the management of “indicators to target” such as blood pressure control, the delayed progression of kidney disease/time to dialysis, and quality of life? What are the costs and costs relative to benefits of the programs/models from the perspectives of individual patients and their families, the primary health services that deliver them, tertiary health services and society as a whole? What do patient and provider experiences of programs/models reveal about the acceptability of programs, as well as barriers and enablers of implementation? Background Chronic kidney disease, and associated chronic illnesses including heart disease, stroke and diabetes, constitutes half of the gap in life expectancy between Indigenous and non-Indigenous Australians.5 Chronic kidney disease occurs more frequently and in younger age groups amongst Aboriginal and Torres Strait Islander people, with rates three to five times the national average in urban areas and up to 30 times the national average in remote areas.11 Mortality rates are correspondingly high with reports from Queensland, South Australia, Western Australia and the Northern Territory listing CKD as a primary or associated cause of death in 16% of Indigenous deaths, a rate at least three and a half times higher than the national average.9 Similarly, a disproportionately high burden of CKD has been found among First Nations people in Canada6,7 and Maori people in New Zealand.8 The scale of the social and economic cost of the progression of CKD to end stage kidney disease (ESKD) in Indigenous Australians is reflected in rates of hospitalization for regular dialysis that are 11 times higher than those recorded for non-Indigenous Australians. Overall, regular dialysis accounts for more than 40% of all hospitalizations for Indigenous Australians.9 Further, the incidence of ESKD in Aboriginal and Torres Strait Islander Australians has more than doubled between 1991 and 2008 and is projected to increase by 130% from 2009 to 2020.10 Dialysis is expensive, invasive and leads to decreased quality of life, particularly for Aboriginal people living in rural and remote locations, who often have to leave their homes for extended periods and/or travel long distances to access treatment.15,16 High rates of clinical and environmental risk factors, including low birth weight, high blood pressure, obesity, smoking, poor nutrition and socioeconomic disadvantage, contribute to the higher burden of CKD in Indigenous populations.10 Reducing this burden will require primary prevention strategies across the life course.11 At present, limited access to appropriate health care in many communities and poor uptake of adult health checks, which partially screen for CKD, present barriers to Aboriginal and Torres Strait Islander Australians accessing timely and appropriate health care for CKD.5 While primary prevention and population based screenings are important health priorities for Indigenous populations as they are for the general population, this review focuses on identifying and synthesizing the evidence on programs and models of care for those who have established CKD but have not reached end-stage, or kidney failure, at which point dialysis or transplant are required (see Figure 1). The goals in management of CKD include the reduction of cardiovascular risk particularly through reducing blood pressure to target levels, early detection and appropriate management of complications, avoidance of nephrotoxic medications, timely referral to a nephrologist, health education and support for diet and other lifestyle changes.12Figure 1: Focus of this review in relation to the prevention and management pathway for CKDFor western, non-Indigenous populations, a recent quantitative systematic review found that care provided by a multidisciplinary team, compared to standard medical care, delays the progression of CKD for adults in the pre-dialysis phase of the condition.13 The four studies included in that review were conducted within the United Kingdom, United States and Canada and focused on education as the primary preventative strategy. While some aspects of the models shown to be effective in non-Indigenous populations may be effective and acceptable for Aboriginal and Torres Strait Islander populations, the evidence suggests that they should not be wholly transferred. For example, the application of self-management approaches to socially disadvantaged populations has been criticized on the basis that they do not take into account the everyday challenges faced by these population groups.14,15 In addition, the individualism of western self-management frameworks sits uncomfortably with the more relational social and cultural context of Indigenous people.16 Further, the ability to access, understand and utilize health information, also known as health literacy, is known to be lower in culturally and linguistically diverse and disadvantaged populations.17 Health literacy is affected by many factors including language barriers, low educational attainment levels, lack of familiarity with medical terminology and differing styles of learning. Research has identified that how and where communication occurs affects how information is received and internalized, with patients clearly preferring settings that align with their worldview. Ideally, health education should result in a shared understanding and involve two way communication, rather than a one way imparting of information.18 This suggests that programs and models of care tailored to the particular needs and context of Indigenous people may be more effective. In order to effectively and appropriately meet the needs of Indigenous people with CKD, programs and models need to fit the social and cultural contexts of Indigenous populations, with a reduced emphasis on the delivery of care within a medical setting. A recent review of cost-effectiveness of diabetes, hypertension and CKD management programs, conducted in the general Australian population, found that primary care-based screening for CKD and its major risk factors, followed by intensive treatment, can lead to improved health outcomes that are also likely to be good value for money.19 The Central Australia Renal Study undertook economic analyses of care provided to Aboriginal people in that region and found that the best value for money would be attained by resourcing prevention efforts to achieve a 20% reduction in the projected rise of ESKD by 2020.20,21,22 A preliminary search of the Joanna Briggs Library of Systematic Reviews, the Cochrane Library, CINAHL, PubMed and PROSPERO revealed that there is not currently a systematic review focused on the proposed topic (either published or underway). This review was conducted as part of a larger collaborative research project designed to meet the need of policy-makers for evidence to inform the building of more effective, efficient and appropriate health care programs and models of care. The mixed method design and questions to be addressed in the review arose from consultation with researchers working on this broader project. This review's findings will be used to highlight the common elements and features of successful programs and provide an evidence informed understanding of key aspects of design and implementation that facilitate success. This review will consider published and unpublished quantitative, economic and qualitative evidence and use the Joanna Briggs Institute mixed method segregated approach for conducting systematic reviews. There will be two distinct phases in the review process. In phase 1 the quantitative, economic and qualitative evidence relevant to the review questions will be identified, assessed and synthesized in a segregated manner, generating three sets of distinct findings. In phase 2, the results from the three segregated components of the review will be drawn together in an aggregative synthesis. Definitions Indigenous: For the purpose of this protocol and systematic review, the term “Indigenous” refers to Aboriginal and Torres Strait Islander Australians, Maori in New Zealand and First Nations people in Canada. Within each of these population groups, there are unique regional and cultural names that are the preferred mode of identifying specific groups. Where possible throughout the systematic review, these names will be used when referring to particular Indigenous subgroup participants of studies. Chronic kidney disease (CKD): This refers to all kidney conditions resulting in kidney damage and/or reduced kidney function, regardless of underlying cause. It is categorized into five stages according to the degree of reduced function. Stage of kidney disease is commonly diagnosed clinically by the estimated glomerular filtration rate (eGFR). This is measured by using a formula requiring age, gender and serum creatinine level in the blood.10 Acceptability: The degree to which a program or model of care is considered acceptable and appropriate by the consumers of care, according to their cultural, social, environmental, geographical, physical and economic needs and preferences. Barriers and facilitators: Any social, economic, cultural, organizational, environmental or personal factor that inhibits or supports access and/or adherence to the health care treatment or program. Effectiveness: The effect of the particular program or model of care on the defined outcomes under “real-world” conditions. This is different from the concept of efficacy, which refers to the effect of a program or model of care under ideal conditions. Cost-effectiveness, cost-benefit and cost-utility analysis: These methods are commonly applied to measure and compare the resource use/costs relative to the benefits/health outcomes/impact of an intervention and comparator. Cost minimization (which assumes benefits are identical for the intervention and comparator) is another method. The approaches are similar (at least in principle) with respect to how they measure cost, but differ in their conceptualization of benefit. The cost benefit approach measures benefits in monetary units, the cost effectiveness approach in natural/clinical outcome units, and the cost utility in quality adjusted life years (QALYS) or disability adjusted life years (DALYS). Outpatient setting: Care provided to people who are not admitted to hospital. It includes outpatient clinics at hospitals, secondary settings, primary healthcare or community settings and includes outreach services to primary health facilities by multidisciplinary and specialist services. Program: For the purpose of this review, a program refers to a health sector led sequence of actions or outline of the way a system or service will function, with specifics such as roles and responsibilities, expected expenditures and outcomes defined. Model of care: a multifaceted concept, which broadly defines the overarching design for the provision of a particular type of health care service. It outlines how healthcare is delivered across clinical streams and patient flow continuums.23–25 Inclusion criteria Types of participants/population Indigenous people (adults 18 years or older) of Australia, Canada and New Zealand diagnosed with CKD AND Receiving care in an outpatient setting. Studies including participants of other ethnicities (or Australian, Canadian and New Zealand populations as a whole), other ages or with additional chronic diseases but where either: the majority of participants match the inclusion criteria; or the results are reported separately for the participants matching the inclusion criteria, will be considered for inclusion. In the qualitative component of the review studies including participants who are Indigenous or non-Indigenous family members, significant others, carers and/or health care providers in Australia, New Zealand and Canada reporting on experiences of health care programs/models matching the inclusion criteria will be considered, in addition to studies whose participants match the above criteria. Types of intervention(s)/phenomena of interest Studies reporting data on health sector led management programs and models of care explicitly designed to manage, slow progression or otherwise improve the lives of people with CKD will be considered for inclusion. Studies evaluating renal replacement therapy (dialysis or transplant) will be excluded. With respect to comparators to be considered in the quantitative effectiveness and economic review components, all health care program/model alternatives will be considered, including comparisons with no CKD management program, usual care, non-Indigenous people or all ethnicities in Australia, New Zealand and Canada. The qualitative component of the review will consider studies that investigate health care worker and/or patient experiences/perceptions of delivery of CKD management programs or models of care to participants matching the inclusion population, in relation to though not limited to, acceptability, patient satisfaction, engagement/participation, self-management and barriers and facilitators of effective CKD management. Context All CKD programs or models of care delivered in the outpatient setting will be considered. Types of outcomes In relation to effectiveness and cost effectiveness, studies will be considered for inclusion if they measure outcomes including, but not limited to: Change in clinical indicators such as HbA1c levels and blood pressure control or evaluate outcomes such as survival and rates of progression to ESKD Quality of life, acceptability and satisfaction Psychosocial and behavioural factors including, but not limited to: ability to self-manage, adherence, depression, anxiety, self-efficacy and service utilization measured with psychometric or other survey instruments Barriers and facilitators to implementation Costs, and/or costs relative to benefits and/or savings associated with implementing the program/model, only implementing part of the model/program, or doing nothing (no CKD program). All measures for the range of included outcomes will be considered and, where relevant, limitations of the measures used for example, when an instrument has not been validated for use with Indigenous populations, will be reported. Types of studies Studies reporting on primary research will be considered for inclusion. Studies to be considered in the element of the review addressing the question of effectiveness are: Randomised controlled trials (RCTs) Non randomised controlled trials Observational studies: Retrospective and prospective cohort studies Case control studies Health service studies Health service evaluations Analytic cross sectional studies Descriptive epidemiological study designs In the component of the review addressing the questions about costs, savings and costs relative to benefits economic evaluations and costing studies (including model based studies) All costing and economic evaluation study designs will be included. Studies based on empirical data only, or empirical data and modelling will be considered. The qualitative review component will consider all qualitative study designs including descriptive, ethnography, phenomenology, grounded theory studies, action research and evaluations including developmental evaluation. If mixed method studies are identified they will be considered for inclusion. Studies that are systematic reviews, solely prevalence studies or epidemiological studies showing relationships between indicators or risk factors in the absence of a specific program or model of care will not be considered for inclusion. Search strategy The search strategy will seek both published and unpublished studies written in English. The date range will be restricted to publications between 2000 and 2014 because technology and data collection in health care has advanced to such an extent that earlier findings are likely to be less relevant in terms of effectiveness, dollar values for cost evaluations and prevailing government strategy, policy and funding arrangements. A three step search strategy will be used. An initial limited search of PubMed and CINAHL will be undertaken followed by analysis of the text words contained in the title and abstract, and of the index terms used to describe the articles. A second search using all identified keywords and index terms will then be undertaken across all included databases. Thirdly, the reference list of all identified reports and articles will be searched for additional studies. The following databases will be searched for published studies: PubMed EBSCO CINAHL Embase ATSIHealth via Informit online Web of Science Psychinfo Social Science Citation Index APAIS Health databases Australian Indigenous Health InfoNet Primary Health Care Research and Information Service (PHCRIS) The search for unpublished studies will include: Mednar and Trove Google Grey OCLC WorldCat Dissertations and Theses Canada Theses Portal Websites of relevant organizations in each country including Kidney Health Australia, Kidney Health New Zealand and The Kidney Foundation of Canada Other specific resources to be searched are: Australian Institute of Torres Strait Islander Studies llt.Search (Lowitja Institute) NativeWeb World Health Organization Hand searching will include Pimatisiwin: Journal of Aboriginal and Indigenous Community Health and reference lists of relevant published systematic reviews. In addition, relevant experts will be consulted. Initial keywords to be used will be: Population of interest (Australia[mh] New New AND Torres Strait First of interest kidney chronic chronic chronic creatinine estimated glomerular filtration disease health rural rural rural health health community delivery of health health health health general of quality quality of studies for will be assessed by two to inclusion in the review using instruments from the Joanna Briggs Institute will be assessed using the Joanna Briggs Institute of and Review will be assessed using the If they include a they will also be using the for will be assessed using the Joanna Briggs Institute and Review Any that between the will be through or with a Stage 1 data economic and qualitative data will be from included in the review using the data from and The data will include specific about the populations, study methods and outcomes of to the review question and specific In addition, a of the data will be to data on elements of programs that are identified as effective in engagement uptake of and satisfaction with health services. will be where the need for to access to publications or information not reported in the methods and Stage data segregated of the included quantitative, economic and qualitative the results of each method included in the mixed method review will be in or For example, for of quantitative this will of appropriate elements of the where an evidence for qualitative reviews, will of appropriate elements of the Stage 1 data for each data where be in using All results will be to data For included studies on the effectiveness of health care programs/models effect as differences and their will be for will be assessed using the standard and also using subgroup analyses based on the different quantitative study designs included in this Where is not possible the findings will be in including and to in data where The evidence on barriers and facilitators will be and synthesized in and data from quantitative will be synthesized using the three by three in and research findings where be using This will involve the or of findings to a of that that through the findings according to their and these findings on the basis of in These are then to a in order to a of synthesized findings that can be used as a basis for Where is not possible the findings will be in Stage data for mixed method The findings of each method included in this review will be This will involve the of the findings to a of that that through quantitative a to all quantitative and all of the resulting from quantitative, qualitative and and the of these to a of synthesized findings in the of a of or the model of mixed method of interest that have no of is by The and Research This project was partially by the Australian of to and for their on the

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