Defining Coordinated Care for People with Rare Conditions: A Scoping Review

Scoping review stage	Description of our method
1) Defined research question	All co-authors developed three research questions: What does coordinated care mean? What are the components of coordinated care? Do definitions and components of care coordination identified in the literature (largely from common chronic conditions) apply to rare conditions?
2) Identified relevant studies	Information sources: – Nine electronic databases (MEDLINE, Scopus, CINAHL Plus, Web of Science, ProQuest Social Science, PubMed, Cochrane Database of systematic reviews, Database of abstracts of reviews of effects, and ProQuest Nursing and Allied Health) (1^stauthor). – One reviewer (1^stauthor) hand-searched key journals (BMJ Quality and Safety, Orphanet Journal of Rare Diseases, Journal of Health Services Research and Policy, Implementation Science). – Searched reference lists of included reviews (1^stauthor). – Sent included reviews to five experts to identify any missing relevant reviews (one responded). Search terms: – Developed around the research questions. – Developed using the search terms used in a review (and the articles citing this review: McDonald et al. [15]), terminology used in coordination grey literature [2, 5, 7; 27, 28, 29] and peer reviewed articles [30, 31, 32] (by 1^st author). – Additional search terms identified through MEDLINE mapping function. – Search terms for reviews based on previous research [33]. – Search strategy reviewed by research team and subject librarian. – Search terms piloted and refined to check identification of key reviews. – Final search conducted (papers published from 2006, up until the date of the search in September 2018). Eligibility criteria (Developed and agreed within the wider research team): Focus on coordination of care within an intervention (Interventions that included both coordinated care and non-coordinated care interventions were excluded. To be inclusive, a range of terms for coordination were included). Focus on chronic or long-term health conditions, including ‘rare’, ‘ultra-rare’, undiagnosed and ‘non-rare’ (common) conditions (To take into account variations in definitions of chronic diseases and rare conditions, broad search terms were used). Provide a definition of coordinated care and information on the components of coordinated care. Review papers (all types of review included as long as a clear method was outlined. For example: narrative reviews, meta-analysis, systematic reviews and scoping reviews). Included health setting (Reviews which included articles that focus on other sectors were also included if they included health setting as well (e.g. social care). Reviews which focused on other sectors alone were excluded) Reviewed international research. Given that rare conditions affect patients all over the world [11], and that different countries have different healthcare systems and variations in how healthcare is delivered, it is important to learn how care is coordinated for both common and rare chronic conditions in different countries with different healthcare systems. Published after 2006 (This year was chosen to capture relevant major policy changes and to take into account a comprehensive review which included reviews prior to 2006 [15]). Written in English Published in peer-reviewed journals or grey literature
3) Selected reviews	– One reviewer (1^st author) conducted search – Guidelines developed around exclusion criteria – Reviewed in three stages: (i) titles, (ii) abstracts, (iii) full texts – Percentage independently screened by 2^nd researcher (2^nd author/TS) ° 40% of titles (n = 712). Agreement for different rounds of title screening ranged from: 60.1–78% ° 30% of abstracts (n = 226). Agreement for different rounds of abstract screening ranged from: 66.7–76% ° 5% of full texts (n = 24). Agreement: 73.9%. Also double screened additional full texts that were unclear (n = 14). – Researchers met to discuss decisions, resolve discrepancies and amend guidelines – One researcher (1st author) checked screening for consistency with amendments – Full-texts that were unclear retained until after data extraction when more information was available [34]
4) Charted data	– Developed a data charting form – One researcher (1^st author) used the form to chart data for all included reviews – Form included: review author, year of publication, review location, details of the programmes reviewed, scope of the review, aims of the review, type of review, outcome measures and important results in relation to the research questions and coordination of care (definitions of coordinated care and components of coordinated care). – Second researcher (2^nd author) extracted data from 10% of reviews identified in the initial electronic and hand search – Researchers met to discuss and resolve discrepancies [23]. – Prior to publication, all data extraction forms were rechecked to ensure the identification of components was comprehensive. Additional information was identified in less than 10% of reviews (n = 14).
5) Collated, summarised and reported results	– Thematic analysis used to develop definitions, identify and group components – 10% of components grouped independently by a second researcher (3^rd author) – Wider research team reviewed and agreed categorisation of components.
6) Consultation with stakeholders	Sample: – Three focus groups were conducted with adults aged 18 or over ° Two focus groups with patients or carers with rare conditions (one virtual focus group (PC1), one face-to-face focus group (PC2)) ° One focus group with healthcare professionals (HCPs) with expertise in rare conditions – Opportunity sampling through our charity partners Procedure: – During the focus groups discussed a summary of early findings from the review ° First focus group (FG-PC1) reviewed findings from 26 reviews ° Second focus group (FG-HCP) reviewed findings from 101 reviews ° Last focus group (FG-PC2) reviewed findings from 127 reviews (all reviews identified prior to expert and reference list searches) – Focus groups were audio-recorded, transcribed and fully anonymised Analysis: – Thematic analysis used to analyse the data – Two researchers inductively coded the three focus group transcripts (1^st/4^th authors) – Findings discussed with research team and refined

Categorisation of components of coordinated care.

A flow chart showing the study selection process (based on Moher et al [52]; PRISMA).

Table 2

Stakeholder consultation participant characteristics.

	Focus group			Total
	1	2	3	Total
Mode of delivery	Virtual	Face-to-face	Face-to-face
Number of participants	7	4	6	17
Type of participant
Patients	4	N/A	2	6
Parents/carers¹	3	N/A	4	7
Healthcare professionals²	N/A	4	N/A	4
Gender
Male	1	0	3	4
Female	6	4	3	13
Age
29–59	6	N/A	3	9
60+	1	N/A	2	3
Not specified	0	N/A	1	1
Diagnosis
One specific rare condition	4	N/A	3	7
Multiple chronic conditions (including at least one rare condition)	2	N/A	3	5
Undiagnosed	1	N/A	0	1
Number of regions³ represented	4	3	4	7

[i] Note: ¹ Parents and carers were included to capture views of caring for adults (n = 2) and children (n = 4) with rare, ultra-rare or undiagnosed conditions.

² Job roles included consultants, nurses and a representative from a rare disease organisation (who had previous experience as a healthcare professional). In addition to their clinical role, one healthcare professional also worked for a rare disease organisation.

³ Regions refers to regions within the United Kingdom, including regions within England, in addition to Scotland, Wales and Northern Ireland.

N/A = not applicable as patients/carers and healthcare professionals were asked different eligibility questions.

A diagram to outline a summary of components of care coordination, from scoping review and stakeholder consultation findings.

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DOI: https://doi.org/10.5334/ijic.5464 | Journal eISSN: 1568-4156

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Language: English

Submitted on: Dec 9, 2019

Accepted on: Jun 1, 2020

Published on: Jun 25, 2020

Published by: Ubiquity Press

In partnership with: Paradigm Publishing Services

Publication frequency: 4 issues per year

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© 2020 Holly Walton, Emma Hudson, Amy Simpson, Angus I. G. Ramsay, Joe Kai, Stephen Morris, Alastair G. Sutcliffe, Naomi J. Fulop, published by Ubiquity Press
This work is licensed under the Creative Commons Attribution 4.0 License.

Volume 20 (2020): Issue 2

Defining Coordinated Care for People with Rare Conditions: A Scoping Review

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