public and patient involvement in health data governance datagov protocol of a CORD-Papers-2022-06-02 (Version 1)

Title: Public and patient involvement in health data governance (DATAGov): protocol of a people-centred mixed-methods study on data use and sharing for rare diseases care and research
Abstract: INTRODUCTION: International policy imperatives for the public and patient involvement in the governance of health data coexist with conflicting cross-border policies on data sharing. This can challenge the planning and implementation of participatory data governance in healthcare services locally. Engaging with local stakeholders and understanding how their needs values and preferences for governing health data can be articulated with policies made at the supranational level is crucial. This paper describes a protocol for a project that aims to coproduce a people-centred model for involving patients and the public in decision-making processes about the use and sharing of health data for rare diseases care and research. METHODS AND ANALYSIS: This multidisciplinary project draws on an explanatory sequential mixed-methods study. A hospital-based survey with patients informal carers health professionals and technical staff recruited at two reference centres for rare diseases in Portugal will be conducted first. The qualitative study will follow consisting of semi-structured interviews and scenario-based workshops with a subsample of the participant groups recruited at baseline. Quantitative data will be analysed using descriptive and inferential statistics. Inductive and deductive approaches will be combined to analyse the qualitative interviews. Data from scenario-based workshops will be iteratively compared using the constant comparison method to identify cross-cutting themes and categories. ETHICS AND DISSEMINATION: The Ethics Committee for Health from the University Hospital Centre So Joo/Faculty of Medicine of University of Porto approved the study protocol (Ref. 99/19). Research findings will be disseminated at academic conferences and science promotion events and through public meetings involving patient representatives practitioners policy-makers and students a project website and peer-reviewed journal publications.
Published: 2021-03-15
Journal: BMJ Open
DOI: 10.1136/bmjopen-2020-044289
DOI_URL: http://doi.org/10.1136/bmjopen-2020-044289
Author Name: de Freitas Cludia
Author link: https://covid19-data.nist.gov/pid/rest/local/author/de_freitas_cludia
Author Name: Amorim Mariana
Author link: https://covid19-data.nist.gov/pid/rest/local/author/amorim_mariana
Author Name: Machado Helena
Author link: https://covid19-data.nist.gov/pid/rest/local/author/machado_helena
Author Name: Leo Teles Elisa
Author link: https://covid19-data.nist.gov/pid/rest/local/author/leo_teles_elisa
Author Name: Baptista Maria Joo
Author link: https://covid19-data.nist.gov/pid/rest/local/author/baptista_maria_joo
Author Name: Renedo Alicia
Author link: https://covid19-data.nist.gov/pid/rest/local/author/renedo_alicia
Author Name: Provoost Veerle
Author link: https://covid19-data.nist.gov/pid/rest/local/author/provoost_veerle
Author Name: Silva Susana
Author link: https://covid19-data.nist.gov/pid/rest/local/author/silva_susana
sha: 004b28323204ed769df765c66beb80727d5f5c48
license: cc-by-nc
license_url: https://creativecommons.org/licenses/by-nc/4.0/
source_x: Medline; PMC
source_x_url: https://www.medline.com/https://www.ncbi.nlm.nih.gov/pubmed/
pubmed_id: 33722870
pubmed_id_url: https://www.ncbi.nlm.nih.gov/pubmed/33722870
pmcid: PMC7959217
pmcid_url: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7959217
url: https://www.ncbi.nlm.nih.gov/pubmed/33722870/ https://doi.org/10.1136/bmjopen-2020-044289
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Keywords Extracted from Text Content: patient patients co-produce leaflets 1 2 3 4 5 6 7 8 9  centre constant-comparison patient Co-produce audio-record friends UHCSJ F o r p e e r 3 employees participants _______________________________________________________________ Faculty Porto c co-produce people DATAGov patients APC COVID-19 Unforeseen ERN EC
Extracted Text Content in Record: First 5000 Characters:Introduction International policy imperatives for public and patient involvement in the governance of health data co-exist with conflicting cross-border policies on data sharing. This can challenge the planning and implementation of participatory data governance in healthcare services locally. Engaging with local stakeholders and understanding how their needs, values and preferences for governing health data can be articulated with policies made at the supranational level is crucial. This paper describes a protocol for a project that aims to co-produce a people-centred model for involving patients and the public in decision-making processes about the use and sharing of health data for rare diseases care and research. This multidisciplinary project draws on an explanatory sequential mixed-methods study. A hospital-based survey with patients, informal carers, health professionals and technical staff recruited at two reference centres for rare diseases in Portugal will be conducted first. The qualitative study will follow consisting of semistructured interviews and scenario-based workshops with a sub-sample of the participant groups recruited at baseline. Quantitative data will be analysed using descriptive and inferential statistics. Inductive and deductive approaches will be combined to analyse the qualitative interviews. Data from scenario-based workshops will be iteratively compared using the constant comparison method to identify cross-cutting themes and categories. 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I confirm all authors consent to publication of this Work and authorise the granting of this licence.  This study will contribute to help reducing health data policy implementation gaps by co-producing a model for public and patient involvement in data governance in the context of rare diseases.  Another of its strengths is its people-centred, multidisciplinary approach designed to elicit stakeholders' expertise to identify challenges to and devise strategies for enacting participatory health data governance.  The project combines the strengths of quantitative and qualitative methods, maximizing the latter's potential to facilitate the engagement of patients and the public with the topic of health data use and sharing.  Although project funds only allow for data collection at two reference centres for rare diseases at one academic hospital centre, this study site was purposefully selected because it would enable access to stakeholders involved in national and international data sharing for research.  Challenges associated with the study's multi-phase design include participant attrition between questionnaires, semi-structured interviews and the scenario-based workshop, and lower response rates, namely by people who may be less resourced to participate or interested in research. How to govern health data has become a question of unprecedented relevance in the golden age of data sharing. 1 Large-scale health data repositories aggregating multiple datasets hold enormous predictive and transformation potential to improve population health and wellbeing. 2 This is especially promising in the field of rare diseases, where transnational data sharing is likely to intensify as a result of increased sponsorship by the European Commission (EC). 3 Yet how health data are shared and used also ca
Keywords Extracted from PMC Text: centre data.41 UHCSJ governance8 patients constant-comparison method50 DATAGov policies.7 27 V.25.0 Porto people's levels46–49 workshops46 constituencies.34 patient used.20 Representatives sensitivity.43 UHCSJ/Faculty leaflets participants ERN governance6 coding,43 powerful-powerless χ2 's COVID-19 coproduce findings.45 Stakeholders' views exclusion).34 37–40
Extracted PMC Text Content in Record: First 5000 Characters:Increasing support for participatory health data governance, whereby patients and other members of the public are involved in consultations and decision making about health data, is premised on the idea that this process can increase accountability in data processing, reduce unnecessary harms and enhance the fair distribution of the benefits of data use.5 6 8 28 Participatory data governance is also encouraged as a way of promoting dialogue about ethical, legal and social issues and imbuing research policy with socioethical sensitivity.29–32 Collaboration between lay and research experts can infuse research with social values and facilitate the codesign of innovative solutions for complex problems.20 31 However, those collaborative relationships are not always easy to develop.33 Careful attention is needed to create participatory spaces that are inclusive and harness everyone's potential for contribution.34–36 Competing interests, and an unbalanced distribution of the resources needed for meaningful participation, can cause disadvantaged groups to be excluded from decision making, which in turn risks to reproduce ethnic, age, gender and socioeconomic inequalities or to even generate new ones (eg, digital exclusion).34 37–40 There is thus an unequivocal need to assess the potential benefits and risks of public and patient involvement in health data governance and to develop a people-centred, bottom-up approach to participation that responds to local stakeholders' interests and needs. This project aims to reach this goal by collaborating with stakeholders engaged in care and research for rare diseases at an academic hospital centre in Portugal. Its specific aims are to: DATAGov is led by a multidisciplinary team working at the crossroads of sociology, public health and clinical medicine and with long-term experience in collaborating with rare diseases community organisations and networks. The project will contribute to advance international policy imperatives for public and patient involvement in health data governance and to improve its practice. It will do so by creating opportunities for stakeholders to discuss the implementation of participatory data governance in a healthcare setting and by eliciting their collective expertise to devise strategies to address emerging ethical, legal and social challenges. This project adopts an explanatory sequential mixed-methods design, collecting and analysing quantitative and qualitative data.41 The quantitative phase is first in the sequence and draws on a hospital-based survey with rare disease patients, informal carers, health professionals and technical staff recruited at the University Hospital Centre São João (UHCSJ), in Porto, Portugal. The qualitative phase relies on semistructured interviews and scenario-based workshops with a subsample of the participants recruited at baseline. Survey and interview data will be collected to examine stakeholders' needs, preferences, expectations and perspectives of public and patient involvement in health data decision making. These findings will be subsequently integrated to develop a set of scenarios for participatory health data governance, which will be presented at the scenario-based workshops. Representatives of all stakeholder groups will be invited to discuss the scenarios and to select a preferred model for involving patients and other members of the public in the governance of health data (see table 1). Integration of quantitative and qualitative findings will provide an understanding of the ethical, legal and social implications of participatory data governance. Data will be collected in two reference centres for rare diseases at the UHCSJ. These research settings were purposefully sampled because: (1) they were among the first reference centres to be created in Portugal in 2016 and are acknowledged for their expertise and clinical practice; (2) they are in charge of overseeing patients from the entire Northern Health Region of Portugal, which allows for larger study samples; (3) one is affiliated with an ERN and the other is awaiting a final decision on its application for membership and (4) they are involved in national and international research projects, which presents an excellent case to explore first-hand experiences of decision making regarding patient data use and sharing. Rare diseases patients, informal carers, health professionals and technical staff will be invited to participate in a survey conducted at the UHCSJ to assess their preferences and needs regarding decision making concerned with health data use and sharing. During a 6-month period, patients aged 12 and above who attend a consultation in the reference centres at the UHCSJ, who can read and write in the Portuguese language and without cognitive impairment will be consecutively recruited to participate in the study, together with their informal carers. Medicine and nursing unit staff of the reference centres outpatient clinics will distribute
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