Public and patient involvement in needs assessment and social innovation: A people-centred approach to care and research for congenital disorders of glycosylation

Cláudia De Freitas, Vanessa Dos Reis, Susana Silva, Paula A. Videira, Eva Morava-Kozicz, Jaak Jaeken

Research output: Contribution to journalArticle

Abstract

Background: Public and patient involvement in the design of people-centred care and research is vital for communities whose needs are underserved, as are people with rare diseases. Innovations devised collectively by patients, caregivers, professionals and other members of the public can foster transformative change toward more responsive services and research. However, attempts to involve lay and professional stakeholders in devising community-framed strategies to address the unmet needs of rare diseases are lacking. In this study, we engaged with the community of Congenital Disorders of Glycosylation (CDG) to assess its needs and elicit social innovations to promote people-centred care and research. Methods: Drawing on a qualitative study, we conducted three think tanks in France with a total of 48 participants, including patients/family members (n = 18), health care professionals (n = 7), researchers (n = 7) and people combining several of these roles (n = 16). Participants came from 20 countries across five continents. They were selected from the registry of the Second World Conference on CDG through heterogeneity and simple random sampling. Inductive and deductive approaches were employed to conduct interpretational analysis using open, axial and selective coding, and the constant-comparison method to facilitate the emergence of categories and core themes. Results: The CDG community has unmet needs for information, quality health care, psychosocial support and representation in decision-making concerned with care and research. According to participants, these needs can be addressed through a range of social innovations, including peer-support communities, web-based information resources and a CDG expertise platform. Conclusion: This is one of the few studies to engage lay and professional experts in needs assessment and innovation for CDG at a global level. Implementing the innovations proposed by the CDG community is likely to have ethical, legal and social implications associated with the potential donation of patients' clinical and biological material that need to be assessed and regulated with involvement from all stakeholders. To promote people-centred care for the CDG community, and increase its participation in the governance of care and research, it is necessary to create participatory spaces in which the views of people affected by CDG can be fully expressed.

Original languageEnglish (US)
Article number682
JournalBMC Health Services Research
Volume17
Issue number1
DOIs
StatePublished - Sep 26 2017
Externally publishedYes

Fingerprint

Congenital Disorders of Glycosylation
Patient Participation
Needs Assessment
Research
Rare Diseases
Quality of Health Care
Caregivers
France
Registries
Decision Making
Research Personnel
Delivery of Health Care

Keywords

  • Congenital disorders of glycosylation
  • ELSI
  • Needs assessment
  • Patient-oriented research
  • People-centred care
  • Public and patient involvement
  • Rare diseases
  • Social innovations

ASJC Scopus subject areas

  • Health Policy

Cite this

Public and patient involvement in needs assessment and social innovation : A people-centred approach to care and research for congenital disorders of glycosylation. / De Freitas, Cláudia; Dos Reis, Vanessa; Silva, Susana; Videira, Paula A.; Morava-Kozicz, Eva; Jaeken, Jaak.

In: BMC Health Services Research, Vol. 17, No. 1, 682, 26.09.2017.

Research output: Contribution to journalArticle

@article{19141a8f4c724fce900c3d5b3fc90f88,
title = "Public and patient involvement in needs assessment and social innovation: A people-centred approach to care and research for congenital disorders of glycosylation",
abstract = "Background: Public and patient involvement in the design of people-centred care and research is vital for communities whose needs are underserved, as are people with rare diseases. Innovations devised collectively by patients, caregivers, professionals and other members of the public can foster transformative change toward more responsive services and research. However, attempts to involve lay and professional stakeholders in devising community-framed strategies to address the unmet needs of rare diseases are lacking. In this study, we engaged with the community of Congenital Disorders of Glycosylation (CDG) to assess its needs and elicit social innovations to promote people-centred care and research. Methods: Drawing on a qualitative study, we conducted three think tanks in France with a total of 48 participants, including patients/family members (n = 18), health care professionals (n = 7), researchers (n = 7) and people combining several of these roles (n = 16). Participants came from 20 countries across five continents. They were selected from the registry of the Second World Conference on CDG through heterogeneity and simple random sampling. Inductive and deductive approaches were employed to conduct interpretational analysis using open, axial and selective coding, and the constant-comparison method to facilitate the emergence of categories and core themes. Results: The CDG community has unmet needs for information, quality health care, psychosocial support and representation in decision-making concerned with care and research. According to participants, these needs can be addressed through a range of social innovations, including peer-support communities, web-based information resources and a CDG expertise platform. Conclusion: This is one of the few studies to engage lay and professional experts in needs assessment and innovation for CDG at a global level. Implementing the innovations proposed by the CDG community is likely to have ethical, legal and social implications associated with the potential donation of patients' clinical and biological material that need to be assessed and regulated with involvement from all stakeholders. To promote people-centred care for the CDG community, and increase its participation in the governance of care and research, it is necessary to create participatory spaces in which the views of people affected by CDG can be fully expressed.",
keywords = "Congenital disorders of glycosylation, ELSI, Needs assessment, Patient-oriented research, People-centred care, Public and patient involvement, Rare diseases, Social innovations",
author = "{De Freitas}, Cl{\'a}udia and {Dos Reis}, Vanessa and Susana Silva and Videira, {Paula A.} and Eva Morava-Kozicz and Jaak Jaeken",
year = "2017",
month = "9",
day = "26",
doi = "10.1186/s12913-017-2625-1",
language = "English (US)",
volume = "17",
journal = "BMC Health Services Research",
issn = "1472-6963",
publisher = "BioMed Central",
number = "1",

}

TY - JOUR

T1 - Public and patient involvement in needs assessment and social innovation

T2 - A people-centred approach to care and research for congenital disorders of glycosylation

AU - De Freitas, Cláudia

AU - Dos Reis, Vanessa

AU - Silva, Susana

AU - Videira, Paula A.

AU - Morava-Kozicz, Eva

AU - Jaeken, Jaak

PY - 2017/9/26

Y1 - 2017/9/26

N2 - Background: Public and patient involvement in the design of people-centred care and research is vital for communities whose needs are underserved, as are people with rare diseases. Innovations devised collectively by patients, caregivers, professionals and other members of the public can foster transformative change toward more responsive services and research. However, attempts to involve lay and professional stakeholders in devising community-framed strategies to address the unmet needs of rare diseases are lacking. In this study, we engaged with the community of Congenital Disorders of Glycosylation (CDG) to assess its needs and elicit social innovations to promote people-centred care and research. Methods: Drawing on a qualitative study, we conducted three think tanks in France with a total of 48 participants, including patients/family members (n = 18), health care professionals (n = 7), researchers (n = 7) and people combining several of these roles (n = 16). Participants came from 20 countries across five continents. They were selected from the registry of the Second World Conference on CDG through heterogeneity and simple random sampling. Inductive and deductive approaches were employed to conduct interpretational analysis using open, axial and selective coding, and the constant-comparison method to facilitate the emergence of categories and core themes. Results: The CDG community has unmet needs for information, quality health care, psychosocial support and representation in decision-making concerned with care and research. According to participants, these needs can be addressed through a range of social innovations, including peer-support communities, web-based information resources and a CDG expertise platform. Conclusion: This is one of the few studies to engage lay and professional experts in needs assessment and innovation for CDG at a global level. Implementing the innovations proposed by the CDG community is likely to have ethical, legal and social implications associated with the potential donation of patients' clinical and biological material that need to be assessed and regulated with involvement from all stakeholders. To promote people-centred care for the CDG community, and increase its participation in the governance of care and research, it is necessary to create participatory spaces in which the views of people affected by CDG can be fully expressed.

AB - Background: Public and patient involvement in the design of people-centred care and research is vital for communities whose needs are underserved, as are people with rare diseases. Innovations devised collectively by patients, caregivers, professionals and other members of the public can foster transformative change toward more responsive services and research. However, attempts to involve lay and professional stakeholders in devising community-framed strategies to address the unmet needs of rare diseases are lacking. In this study, we engaged with the community of Congenital Disorders of Glycosylation (CDG) to assess its needs and elicit social innovations to promote people-centred care and research. Methods: Drawing on a qualitative study, we conducted three think tanks in France with a total of 48 participants, including patients/family members (n = 18), health care professionals (n = 7), researchers (n = 7) and people combining several of these roles (n = 16). Participants came from 20 countries across five continents. They were selected from the registry of the Second World Conference on CDG through heterogeneity and simple random sampling. Inductive and deductive approaches were employed to conduct interpretational analysis using open, axial and selective coding, and the constant-comparison method to facilitate the emergence of categories and core themes. Results: The CDG community has unmet needs for information, quality health care, psychosocial support and representation in decision-making concerned with care and research. According to participants, these needs can be addressed through a range of social innovations, including peer-support communities, web-based information resources and a CDG expertise platform. Conclusion: This is one of the few studies to engage lay and professional experts in needs assessment and innovation for CDG at a global level. Implementing the innovations proposed by the CDG community is likely to have ethical, legal and social implications associated with the potential donation of patients' clinical and biological material that need to be assessed and regulated with involvement from all stakeholders. To promote people-centred care for the CDG community, and increase its participation in the governance of care and research, it is necessary to create participatory spaces in which the views of people affected by CDG can be fully expressed.

KW - Congenital disorders of glycosylation

KW - ELSI

KW - Needs assessment

KW - Patient-oriented research

KW - People-centred care

KW - Public and patient involvement

KW - Rare diseases

KW - Social innovations

UR - http://www.scopus.com/inward/record.url?scp=85029868729&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85029868729&partnerID=8YFLogxK

U2 - 10.1186/s12913-017-2625-1

DO - 10.1186/s12913-017-2625-1

M3 - Article

C2 - 28950866

AN - SCOPUS:85029868729

VL - 17

JO - BMC Health Services Research

JF - BMC Health Services Research

SN - 1472-6963

IS - 1

M1 - 682

ER -