Evidence map›Paper›PMID 35089945›Full record

Trial reportPloS one2022

Assessing women's preferences towards tests that may reveal uncertain results from prenatal genomic testing: Development of attributes for a discrete choice experiment, using a mixed-methods design.

Jennifer Hammond, Jasmijn E Klapwijk, Sam Riedijk, Stina Lou, Kelly E Ormond, Ida Vogel, Lisa Hui, Emma-Jane Sziepe, James Buchanan, Charlotta Ingvoldstad-Malmgren and 4 more

Abstract readClinical TrialMulticenter Study
In one paragraph

Trial report in PloS one, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.

0numbers the graph read from it
0cells of the map it votes in
12citing 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

12 citing papers in PubMed.

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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

14 authors.

Jennifer HammondNorth Thames Genomic Laboratory Hub, Great Ormond Street Hospital, London, United Kingdom.
Jasmijn E KlapwijkDepartment of Clinical Genetics, Erasmus MC, Rotterdam, The Netherlands.ORCID 0000-0002-6775-6561
Sam RiedijkDepartment of Clinical Genetics, Erasmus MC, Rotterdam, The Netherlands.
Stina LouCenter for Fetal Diagnostics, Department of Clinical Medicine, Aarhus University, Aarhus, Denmark.
Kelly E OrmondDepartment of Genetics and Stanford Center for Biomedical Ethics, Stanford University School of Medicine, Stanford, CA, United States America.ORCID 0000-0002-1033-0818
Ida VogelCenter for Fetal Diagnostics, Department of Clinical Medicine, Aarhus University, Aarhus, Denmark.
Lisa HuiDepartment of Obstetrics and Gynaecology, University of Melbourne, Parkville, Victoria, Australia.
Emma-Jane SziepeReproductive Epidemiology, Murdoch Children's Research Institute, Royal Children's Hospital, Parkville, Victoria, Australia.
James BuchananHealth Economics Research Centre, Nuffield Department of Population Health, University of Oxford, Oxford, England, United Kindom.
Charlotta Ingvoldstad-MalmgrenDepartment of Clinical Genetics, Karolinska Hospital and Karolinska Institutet, Stockholm, Sweden.
Maria Johansson SollerDepartment of Clinical Genetics, Karolinska Hospital and Karolinska Institutet, Stockholm, Sweden.
Eleanor HardingBSc Paediatrics and Child Health, The UCL Great Ormond Street Institute of Child Health, London, United Kingdom.
Melissa HillNorth Thames Genomic Laboratory Hub, Great Ormond Street Hospital, London, United Kingdom.
Celine LewisNorth Thames Genomic Laboratory Hub, Great Ormond Street Hospital, London, United Kingdom.ORCID 0000-0001-7169-1521

Funding

Wellcome Trust
6 · The paper itself

Abstract

Prenatal DNA tests, such as chromosomal microarray analysis or exome sequencing, increase the likelihood of receiving a diagnosis when fetal structural anomalies are identified. However, some parents will receive uncertain results such as variants of uncertain significance and secondary findings. We aimed to develop a set of attributes and associated levels for a discrete-choice experiment (DCE) that will examine parents' preferences for tests that may reveal uncertain test results. A two phase mixed-methods approach was used to develop attributes for the DCE. In Phase 1, a "long list" of candidate attributes were identified via two approaches: 1) a systematic review of the literature around parental experiences of uncertainty following prenatal testing; 2) 16 semi-structured interviews with parents who had experienced uncertainty during pregnancy and 25 health professionals who return uncertain prenatal results. In Phase 2, a quantitative scoring exercise with parents prioritised the candidate attributes. Clinically appropriate levels for each attribute were then developed. A final set of five attributes and levels were identified: likelihood of getting a result, reporting of variants of uncertain significance, reporting of secondary findings, time taken to receive results, and who tells you about your result. These attributes will be used in an international DCE study to investigate preferences and differences across countries. This research will inform best practice for professionals supporting parents to manage uncertainty in the prenatal setting.

Indexed as

Genetic TestingGenomicsPatient PreferencePrenatal DiagnosisAdultFemaleHumansMiddle Aged

Identifiers

PMID35089945
PMCPMC8797177

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.