Children’s, parents’ and other stakeholders’ perspectives on early dietary self-management to delay disease progression of chronic disease in children: a protocol for a mixed studies systematic review with a narrative synthesis

The aim of this study is to provide decision-makers with perspectives on early dietary intervention, as a form of self-management, to delay disease progression in children with early chronic disease, as described by children, parents and other stakeholders. This will provide insight into the current thinking on early dietary self-management in children with chronic disease, and how this relates to early chronic kidney disease stages 1–3, in order to provide a strategy for service development. By its nature, the exploration of multiple stakeholder perspectives makes the focus of this review complex. A mixed studies approach, which appeals for a concomitant examination of qualitative, quantitative and mixed methods primary studies, will address the broad purpose of the scope, understanding and verification of knowledge grounded on all types of empirical research. A Mixed Studies Review with a narrative synthesis is a literature review approach in which the narrative element of qualitative, quantitative and mixed methods studies is systematically identified, selected, appraised and synthesised. Due to the complex and highly context-sensitive nature of interventions, a mixed studies review is particularly relevant to health science [31, 32]. A mixed studies review can provide a better understanding of a health issue than when one type of research approach is used alone. This mixed studies review will have an exploratory purpose where the qualitative component dominates. The use of Pluye et al.’s eight-stage Question Eligibility Source Identification Selection Appraisal Extract and Synthesis (QESISAES) framework [33], which is in line with the PRISMA statement (www.​prisma-statement.​org), will enable us to (1) to identify, appraise and synthesise qualitative, quantitative and mixed methods design empirical research; (2) to identify the facilitators and barriers to providing early dietary self-management for children with chronic disease and CKD 1–3; and (3) to disseminate the findings strategically via a managed paediatric renal network to influence commissioners and government level decisions. The review has been registered with PROSPERO (CRD42017078130).

The PICO tool was used to define the research question (Table 2).

The research question is as follows: What are the views of children, parents’ and others stakeholders’ towards the use of an early dietary intervention as a form of self-management, to delay disease progression, for children aged 0–16 years with an early chronic disease?

The outcome of the study is to delay the progression of the chronic disease.

The search will utilise established systematic review methodologies as detailed in the Centre for Reviews and Dissemination Guide for Undertaking Reviews in Health Care [37] and integrate guidelines for the selection, appraisal and review of the grey literature [38, 39].

Health service researchers regularly produce and use grey literature to defend their inquiry. Chronic care and chronic care delivery are priority subject areas [39] for grey literature.

This Mixed Studies Review search strategy will cross multiple databases as it seeks to identify both published and unpublished studies using a four-stage approach. In order to retrieve maximum coverage of the literature, careful thought was given to the most appropriate databases to search. (1) An initial scoping search of MEDLINE, CINAHL, Cochrane, EMBASE, Scopus, PsycINFO and OpenGrey failed to identify sufficient literature on early dietary self-management in children with CKD. However, given the importance of diet in the management and prevention of progressive chronic disease, the findings suggested there would be sufficient literature to inform a systematic review if the search was extended to all chronic disease in childhood. (2) The Scottish Intercollegiate Guidelines Network (SIGN) (website www.​sign.​ac.​uk) has a validated Patient Issues search filters; this was applied to MEDLINE. Despite providing a broad coverage of patient issues, it did not deliver the required specificity to address the research question. With the support of an information scientist (health research librarian), an analysis of the search terms and Medical Search Headings (MeSH) contained in the title and abstract was used to write a chronic disease and CKD search strategy to reflect the review question. (3) This search strategy was applied to seven databases (see Table 3). (4) Finally, the reference list of all identified papers will be hand searched for additional studies. Author searches and contact will be performed as necessary.

Chronic Disease OR Chronic Illness OR Chronic Disorder OR Chronic Kidney Disease OR Chronic Kidney Failure OR Renal Insufficiency OR CKD; AND Patient OR Adolescent OR Child; AND Parent OR Carer OR Nurse OR Consultant OR Physician OR Dietitian OR Allied Health Personnel; AND Self-management OR Self-care OR Early intervention OR Secondary prevention; AND Disease progression OR Time OR Endpoint; AND Diet OR Nutrition OR Diet therapy OR Food; AND Perspectives OR Views OR Concern OR Attitude.

A detailed search strategy for MEDLINE is shown in Additional file 1.

Using the criteria for inclusion (Table 4), relevant literature will be identified for further scrutiny.

Studies will be imported into EndNote X7 reference manager. Duplicates will be excluded, and remaining studies will be subjected to a title, abstract and full-text sift against the inclusion/exclusion criteria. Studies that state lifestyle changes, without mention of a dietary component, will be excluded due to ambiguity.

All descriptive and analytic research study designs will be included [40]. Therefore, reviews, editorials, commentaries and expert opinion papers will be excluded, although the reference list of reviews will be searched for relevant literature. A PRISMA flow diagram will present the results of the search process [41] (Fig. 1).

Tyndall’s checklist (Additional file 2) will be used to appraise the quality and relative importance of grey literature [42]. The checklist will appraise the following aspect of grey literature: authority, accuracy, coverage, objectivity, date and significance.

Grey literature that passes the appraisal must present a clear research question(s), with key findings, and give adequate details on the population studied, interventions, and study design, a method of analysis and evaluation outcomes [38].

All relevant studies retrieved through the search strategy will be appraised for methodological quality by a primary and secondary reviewer before inclusion in the study. The Mixed Methods Appraisal Tool (MMAT) [43, 44] and scoring system (Additional file 3) will be used. It is an efficient and validated tool [45] (Additional file 4), and the only available tool that allows the concomitant quality appraisal of qualitative, quantitative and mixed methods studies (mixed studies reviews). Any disagreements that arise between the reviewers will be resolved through discussion, achieving consensus with a third reviewer if necessary.

A relevancy appraisal will be undertaken by the primary author by first reviewing the title and abstract. Potentially relevant articles which match the inclusion criteria will be retrieved, and the whole team (PP, PH, MC, GH) will decide on the final inclusion. Data extraction will primarily be undertaken by one researcher (PP); however, the whole team will meet to decide on which data variables to include in the data extraction checklist. The type of data to be extracted will be determined by the nature of the studies, with a focus on how best to answer the research question. The Centre for Review and Dissemination guidance [37] and Popay et al.’s work [46] provide a suggested focus for the data extraction. An example of the type of data that will be extracted is detailed in Table 5.

Narrative synthesis is a procedure for describing, comparing and combining heterogeneous qualitative findings and quantitative results using text and illustrations [47]. The synthesis will use elements of Popay et al.’s ([46], p. 5) methodology of narrative synthesis, which involves ‘telling a trustworthy story’. It is defined as an ‘approach to the systematic review and synthesis of findings from multiple studies that rely primarily on the use of words and text to summarise and explain the findings of the synthesis’ [46] (p. 5). Both qualitative, quantitative and mixed methods studies can utilise this approach, as the focus is on the interpretive synthesis of the narrative findings of the research as opposed to a meta-analysis of the data. This approach will provide access to the multi-disciplinary perspective of stakeholders and provide a dynamic viewpoint for evaluating the plurality of health-related knowledge on early dietary self-management in chronic diseases, using multiple methodologies.

A narrative synthesis comprises four key elements:

We will follow the steps outlined by Popay et al. [46].

The narrative synthesis brings on a cyclical process with an interweaving of the key elements. Within each element, various tools will be employed to suit the nature of the evidence. Many of these tools will assess the risk of bias.

It is our aim to apply the findings of the review to the management of CKD in childhood, with the anticipation that other relevant paediatric specialities will do likewise. The main findings of the review will be disseminated to key stakeholders across a managed paediatric nephrology network, in order to impact policy and practice change in early paediatric nephrology service delivery. The managed network has an online resource hub for professionals, children and carers which would serve as a powerful dissemination vehicle. Likewise, the network holds bi-annual multi-professional team education and training meetings where the findings can be publicised. Publication of the findings in accessible health science journals will make a contribution to academic theory and practice. Also, as the lead author is engaged in clinical practice with children with early CKD and their families, face to face dissemination will be ongoing.