Defining core outcomes of reproductive genetic carrier screening: A Delphi survey of Australian and New Zealand stakeholders.

Richardson, E; McEwen, A; Newton-John, T; Jacobs, C

Defining core outcomes of reproductive genetic carrier screening: A Delphi survey of Australian and New Zealand stakeholders.

Richardson, E McEwen, A

Newton-John, T

Jacobs, C

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Publisher:: Wiley
Publication Type:: Journal Article
Citation:: Prenat Diagn, 2023, 43, (9), pp. 1150-1165
Issue Date:: 2023-08

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Field	Value	Language
dc.contributor.author	Richardson, E
dc.contributor.author	McEwen, A https://orcid.org/0000-0001-8705-1190
dc.contributor.author	Newton-John, T https://orcid.org/0000-0003-4219-4985
dc.contributor.author	Jacobs, C https://orcid.org/0000-0002-9557-9080
dc.date.accessioned	2023-09-05T06:08:20Z
dc.date.available	2023-07-17
dc.date.available	2023-09-05T06:08:20Z
dc.date.issued	2023-08
dc.identifier.citation	Prenat Diagn, 2023, 43, (9), pp. 1150-1165
dc.identifier.issn	0197-3851
dc.identifier.issn	1097-0223
dc.identifier.uri	http://hdl.handle.net/10453/171920
dc.description.abstract	OBJECTIVE: Understanding the value, benefits and harms of health interventions is needed to inform best practice and ensure responsible implementation of new approaches to patient care. Such value is demonstrated through the assessment of outcomes; however, which outcomes are assessed is often highly varied across studies and can hinder the ability to draw robust conclusions. The Core Outcome Development for Carrier Screening study aims to understand the outcomes that can meaningfully capture the value of reproductive genetic carrier screening (RGCS). METHOD: The authors report an iterative, two-round online Delphi survey of Australian and New Zealand stakeholders to determine the degree of consensus regarding the core outcomes of RGCS. Panellists ranked 83 outcomes according to their perceived importance on a nine-point Likert scale. Using the distribution of rankings, outcomes were grouped into tiers representative of their perceived level of importance and agreement between groups. RESULTS: The top tier outcomes represent those agreed to be critically important for all future studies of RGCS to assess and were used to define a preliminary core outcome set encompassing the domains (1) primary laboratory outcomes, (2) pregnancy outcomes, (3) resource use and, (4) perceived utility of RGCS. CONCLUSION: These findings can guide the selection of meaningful outcomes in studies aiming to demonstrate the value of RGCS. A future international consensus process will expand on these findings and guide the inclusion of diverse perspectives across the range of settings in which RGCS is offered.
dc.format	Print-Electronic
dc.language	eng
dc.publisher	Wiley
dc.relation.ispartof	Prenat Diagn
dc.relation.isbasedon	10.1002/pd.6410
dc.rights	info:eu-repo/semantics/openAccess
dc.subject	1103 Clinical Sciences, 1114 Paediatrics and Reproductive Medicine
dc.subject.classification	Obstetrics & Reproductive Medicine
dc.subject.classification	3202 Clinical sciences
dc.subject.classification	3215 Reproductive medicine
dc.subject.mesh	Pregnancy
dc.subject.mesh	Female
dc.subject.mesh	Humans
dc.subject.mesh	Genetic Carrier Screening
dc.subject.mesh	New Zealand
dc.subject.mesh	Delphi Technique
dc.subject.mesh	Australia
dc.subject.mesh	Research Design
dc.subject.mesh	Outcome Assessment, Health Care
dc.subject.mesh	Humans
dc.subject.mesh	Pregnancy
dc.subject.mesh	Research Design
dc.subject.mesh	Delphi Technique
dc.subject.mesh	Australia
dc.subject.mesh	New Zealand
dc.subject.mesh	Female
dc.subject.mesh	Genetic Carrier Screening
dc.subject.mesh	Outcome Assessment, Health Care
dc.title	Defining core outcomes of reproductive genetic carrier screening: A Delphi survey of Australian and New Zealand stakeholders.
dc.type	Journal Article
utslib.citation.volume	43
utslib.location.activity	England
utslib.for	1103 Clinical Sciences
utslib.for	1114 Paediatrics and Reproductive Medicine
pubs.organisational-group	/University of Technology Sydney
pubs.organisational-group	/University of Technology Sydney/Faculty of Health
pubs.organisational-group	/University of Technology Sydney/Strength - CHT - Health Technologies
pubs.organisational-group	/University of Technology Sydney/Faculty of Health/Graduate School of Health
pubs.organisational-group	/University of Technology Sydney/Faculty of Health/Graduate School of Health/GSH.Genetic Counselling
pubs.organisational-group	/University of Technology Sydney/Centre for Health Technologies (CHT)
utslib.copyright.status	open_access	*
dc.date.updated	2023-09-05T06:08:19Z
pubs.issue	9
pubs.publication-status	Published
pubs.volume	43
utslib.citation.issue	9

Abstract:

OBJECTIVE: Understanding the value, benefits and harms of health interventions is needed to inform best practice and ensure responsible implementation of new approaches to patient care. Such value is demonstrated through the assessment of outcomes; however, which outcomes are assessed is often highly varied across studies and can hinder the ability to draw robust conclusions. The Core Outcome Development for Carrier Screening study aims to understand the outcomes that can meaningfully capture the value of reproductive genetic carrier screening (RGCS). METHOD: The authors report an iterative, two-round online Delphi survey of Australian and New Zealand stakeholders to determine the degree of consensus regarding the core outcomes of RGCS. Panellists ranked 83 outcomes according to their perceived importance on a nine-point Likert scale. Using the distribution of rankings, outcomes were grouped into tiers representative of their perceived level of importance and agreement between groups. RESULTS: The top tier outcomes represent those agreed to be critically important for all future studies of RGCS to assess and were used to define a preliminary core outcome set encompassing the domains (1) primary laboratory outcomes, (2) pregnancy outcomes, (3) resource use and, (4) perceived utility of RGCS. CONCLUSION: These findings can guide the selection of meaningful outcomes in studies aiming to demonstrate the value of RGCS. A future international consensus process will expand on these findings and guide the inclusion of diverse perspectives across the range of settings in which RGCS is offered.

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http://hdl.handle.net/10453/171920