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A validated PROM in genetic counselling: the psychometric properties of the Dutch version of the Genetic Counselling Outcome Scale

A validated PROM in genetic counselling: the psychometric properties of the Dutch version of the Genetic Counselling Outcome Scale
A validated PROM in genetic counselling: the psychometric properties of the Dutch version of the Genetic Counselling Outcome Scale
Patient empowerment has been identified as a key outcome goal in genetic counselling, and a patient reported outcome measure (PROM) has been developed to measure empowerment in genetic services: the Genetic Counselling Outcome Scale (GCOS). Here we validate the GCOS for a large and diverse Dutch study sample of 2194 patients referred to two clinical genetic centres for counselling about a wide range of conditions (heart disease, neurological disorders, cancer, congenital syndromes, intellectual disability and prenatal pathology). Our results suggest that the GCOS consists of a hierarchical 6-factor structure, with a main scale for empowerment and six subscales: uncertainty about heredity, hope, negative emotions, knowledge about the condition, knowledge about genetic services and uncertainty about the treatment. Six of the original 24 GCOS items were removed due to low factor loadings and small inter-item correlations. Internal consistency and test-retest reliability of the main scale and most subscales were satisfactory. Convergent validity was confirmed by moderate positive and moderate/strong negative associations between the GCOS main scale and other validated outcome measures. Responsiveness was comparable to that of other validated outcome measures. We saw significant improvement in the GCOS main scale and all the subscales after the first genetic counselling session. This study contributes to the international validation process of the GCOS, with the ultimate goal of using this instrument as a PROM, with empowerment as an outcome measure, to evaluate and improve the quality of genetic counselling in various clinical genetics settings.
1018-4813
681-690
Krijnen, Wrm
f10010b1-0e68-4eb3-a085-2fe91bf89ef8
Ausems, Margreet G.E.M.
25eb17d5-51dc-4aae-9c60-1a57f67bb648
Knoers, Nine V.A.M.
bbca3ab7-b855-4287-9055-54cf0c082741
Velthuizen, Mary
4c919221-c67c-43bc-8a09-887fd23db38a
Birnie, Erwin
dad5871f-46ff-4765-9c3f-b2d89aa71a31
Van Langen, Irene M.
bcc17f78-800f-4deb-ac58-7c7e2f408bc7
Lucassen, Anneke
2eb85efc-c6e8-4c3f-b963-0290f6c038a5
Ranchor, Adelita V.
6097efc1-d863-4171-bc44-16f01cf6ab91
Krijnen, Wrm
f10010b1-0e68-4eb3-a085-2fe91bf89ef8
Ausems, Margreet G.E.M.
25eb17d5-51dc-4aae-9c60-1a57f67bb648
Knoers, Nine V.A.M.
bbca3ab7-b855-4287-9055-54cf0c082741
Velthuizen, Mary
4c919221-c67c-43bc-8a09-887fd23db38a
Birnie, Erwin
dad5871f-46ff-4765-9c3f-b2d89aa71a31
Van Langen, Irene M.
bcc17f78-800f-4deb-ac58-7c7e2f408bc7
Lucassen, Anneke
2eb85efc-c6e8-4c3f-b963-0290f6c038a5
Ranchor, Adelita V.
6097efc1-d863-4171-bc44-16f01cf6ab91

Krijnen, Wrm, Ausems, Margreet G.E.M., Knoers, Nine V.A.M., Velthuizen, Mary, Birnie, Erwin, Van Langen, Irene M., Lucassen, Anneke and Ranchor, Adelita V. (2019) A validated PROM in genetic counselling: the psychometric properties of the Dutch version of the Genetic Counselling Outcome Scale. European Journal of Human Genetics, 27, 681-690. (doi:10.1038/s41431-018-0318-9).

Record type: Article

Abstract

Patient empowerment has been identified as a key outcome goal in genetic counselling, and a patient reported outcome measure (PROM) has been developed to measure empowerment in genetic services: the Genetic Counselling Outcome Scale (GCOS). Here we validate the GCOS for a large and diverse Dutch study sample of 2194 patients referred to two clinical genetic centres for counselling about a wide range of conditions (heart disease, neurological disorders, cancer, congenital syndromes, intellectual disability and prenatal pathology). Our results suggest that the GCOS consists of a hierarchical 6-factor structure, with a main scale for empowerment and six subscales: uncertainty about heredity, hope, negative emotions, knowledge about the condition, knowledge about genetic services and uncertainty about the treatment. Six of the original 24 GCOS items were removed due to low factor loadings and small inter-item correlations. Internal consistency and test-retest reliability of the main scale and most subscales were satisfactory. Convergent validity was confirmed by moderate positive and moderate/strong negative associations between the GCOS main scale and other validated outcome measures. Responsiveness was comparable to that of other validated outcome measures. We saw significant improvement in the GCOS main scale and all the subscales after the first genetic counselling session. This study contributes to the international validation process of the GCOS, with the ultimate goal of using this instrument as a PROM, with empowerment as an outcome measure, to evaluate and improve the quality of genetic counselling in various clinical genetics settings.

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More information

Accepted/In Press date: 4 December 2018
e-pub ahead of print date: 25 January 2019
Published date: May 2019

Identifiers

Local EPrints ID: 430400
URI: http://eprints.soton.ac.uk/id/eprint/430400
ISSN: 1018-4813
PURE UUID: dbeb65dd-5f04-422b-bf37-400701d28692
ORCID for Anneke Lucassen: ORCID iD orcid.org/0000-0003-3324-4338

Catalogue record

Date deposited: 26 Apr 2019 16:30
Last modified: 26 Nov 2021 02:45

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Contributors

Author: Wrm Krijnen
Author: Margreet G.E.M. Ausems
Author: Nine V.A.M. Knoers
Author: Mary Velthuizen
Author: Erwin Birnie
Author: Irene M. Van Langen
Author: Anneke Lucassen ORCID iD
Author: Adelita V. Ranchor

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