Development of a measure of genome sequencing knowledge for young people: The kids-KOGS.


Journal

Clinical genetics
ISSN: 1399-0004
Titre abrégé: Clin Genet
Pays: Denmark
ID NLM: 0253664

Informations de publication

Date de publication:
11 2019
Historique:
received: 01 04 2019
revised: 10 07 2019
accepted: 16 07 2019
pubmed: 20 7 2019
medline: 1 9 2020
entrez: 20 7 2019
Statut: ppublish

Résumé

Genome sequencing (GS) is increasingly being used to diagnose rare diseases in paediatric patients; however, no measures exist to evaluate their knowledge of this technology. We aimed to develop a robust measure of knowledge of GS (the kids-KOGS') suitable for use in the paediatric setting as well as for general public education. The target age was 11 to 15 year olds. An iterative process involving six sequential stages was conducted to develop a set of draft true/false items. These were then administered to 539 target-age school pupils (mean 12.8; SD ± 1.3), from the United Kingdom. Item-response theory was used to confirm the psychometric suitability of the candidate items. None of the Items was identified as misfits. All 10 items performed well under the two-parameter logistic model. The internal consistency of the test was 0.84 (Cronbach alpha value) indicating excellent reliability. The mean kids-KOGS score in the sample overall was 4.24 (SD; 2.49), where 0 = low knowledge and 10 = high knowledge. Age was positively associated with score in a multivariate linear regression. The kids-KOGS is a short and reliable tool that can be used by researchers and healthcare professionals offering GS to paediatric patients. Further validation in a clinical setting is required.

Identifiants

pubmed: 31323115
doi: 10.1111/cge.13607
pmc: PMC6851564
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

411-417

Subventions

Organisme : Department of Health
ID : PB-PG-1014-35016
Pays : United Kingdom

Informations de copyright

© 2019 The Authors. Clinical Genetics published by John Wiley & Sons Ltd.

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Auteurs

Celine Lewis (C)

North East Thames Regional Genetics Services, Great Ormond Street Hospital NHS Foundation Trust, London, UK.
UCL Great Ormond Street Institute of Child Health, London, UK.

Bao S Loe (BS)

The Psychometrics Centre, University of Cambridge, Cambridge, UK.

Chris Sidey-Gibbons (C)

Patient Reported Outcomes, Value and Experience (PROVE) Centre, Brigham and Women's Hospital, Boston, Massachusetts.

Christine Patch (C)

Genomics England, Queen Mary University of London, London, UK.
Counselling, Society and Ethics Research, Wellcome Genome Campus, Cambridge, UK.
Florence Nightingale Faculty of Nursing and Midwifery, King's College London, London, UK.

Lyn S Chitty (LS)

North East Thames Regional Genetics Services, Great Ormond Street Hospital NHS Foundation Trust, London, UK.
UCL Great Ormond Street Institute of Child Health, London, UK.

Saskia C Sanderson (SC)

North East Thames Regional Genetics Services, Great Ormond Street Hospital NHS Foundation Trust, London, UK.
Institute of Health Informatics, University College London, London, UK.

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Classifications MeSH