Evidence map›Paper›PMID 41917524›Full record

ArticleJournal of patient-reported outcomes2026

A framework to amplify the voices of underrepresented children and young people in rare disease research.

L Thomas, J Preston, H Hardwick, J D Peipert, L Oni

Abstract read
In one paragraph

Article in Journal of patient-reported outcomes, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

5 authors.

L ThomasDepartment of Women's and Children's Health, University of Liverpool, Liverpool, UK.
J PrestonDepartment of Women's and Children's Health, University of Liverpool, Liverpool, UK.
H HardwickDepartment of Women's and Children's Health, University of Liverpool, Liverpool, UK.
J D PeipertCentre for Patient Reported Outcomes Research, Department of Applied Health Sciences, University of Birmingham, Birmingham, UK.
L OniDepartment of Women's and Children's Health, University of Liverpool, Liverpool, UK. louise.oni@liverpool.ac.uk.ORCID http://orcid.org/0000-0002-1532-2390

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Rare diseases predominantly affect children and young people (CYP). Current research involving CYP often fails to adequately represent those from lower socioeconomic backgrounds, contributing to widening health and research inequalities. This concise report proposes a framework to build on successful methods that were able to engage with underrepresented CYP. This was achieved through partnership with social enterprise organisations and utilised connections in the community to provide positive knowledge exchange of local services that may enhance their socioeconomic situation, in turn for engaging on how to improve services or advance research. These insights, together with information from scoping the field, were used to develop a practical framework for inclusive Patient and Public Involvement and Engagement (PPIE) with CYP. This framework has been initially designed to be evaluated in CYP with rare kidney diseases, as an exemplar long term health condition. The model of ‘PPIE through collaborative empowerment’ leverages existing community trust to reduce barriers to participation, hopefully opening the door to more representative views from children residing in regions of low socioeconomic status. The framework has potential to be adapted to address other inequalities or adaptation for different diseases, hopefully offering a replicable approach to representative research design. The next stages of this research are to implement the framework and formally evaluate its success.

Indexed as

Biomedical ResearchMinority GroupsRare DiseasesAdolescentChildHumansPatient ParticipationSocioeconomic Disparities in Health

Identifiers

PMID41917524
PMCPMC13161448

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.