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A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial

INTRODUCTION: In current practice, probands are asked to inform relatives about the possibility of predictive DNA testing when a pathogenic variant causing an inherited cardiac condition (ICC) is identified. Previous research on the uptake of genetic counselling and predictive DNA testing in relativ...

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Autores principales: van den Heuvel, Lieke M, Hoedemaekers, Yvonne M, Baas, Annette F, van Tintelen, J Peter, Smets, Ellen M A, Christiaans, Imke
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BMJ Publishing Group 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6615798/
https://www.ncbi.nlm.nih.gov/pubmed/31289060
http://dx.doi.org/10.1136/bmjopen-2018-025660
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author van den Heuvel, Lieke M
Hoedemaekers, Yvonne M
Baas, Annette F
van Tintelen, J Peter
Smets, Ellen M A
Christiaans, Imke
author_facet van den Heuvel, Lieke M
Hoedemaekers, Yvonne M
Baas, Annette F
van Tintelen, J Peter
Smets, Ellen M A
Christiaans, Imke
author_sort van den Heuvel, Lieke M
collection PubMed
description INTRODUCTION: In current practice, probands are asked to inform relatives about the possibility of predictive DNA testing when a pathogenic variant causing an inherited cardiac condition (ICC) is identified. Previous research on the uptake of genetic counselling and predictive DNA testing in relatives suggests that not all relatives are sufficiently informed. We developed a randomised controlled trial to evaluate the effectiveness of a tailored approach in which probands decide together with the genetic counsellor which relatives they inform themselves and which relatives they prefer to have informed by the genetic counsellor. Here, we present the study protocol of this randomised controlled trial. METHODS: A multicentre randomised controlled trial with parallel-group design will be conducted in which an intervention group receiving the tailored approach will be compared with a control group receiving usual care. Adult probands diagnosed with an ICC in whom a likely pathogenic or pathogenic variant is identified will be randomly assigned to the intervention or control group (total sample: n=85 probands). Primary outcomes are uptake of genetic counselling and predictive DNA testing by relatives (total sample: n=340 relatives). Secondary outcomes are appreciation of the approach used and impact on familial and psychological functioning, which will be assessed using questionnaires. Relatives who attend genetic counselling will be asked to fill out a questionnaire as well. ETHICS AND DISSEMINATION: Ethical approval was obtained from the Medical Ethical Committee of the Amsterdam University Medical Centres (MEC 2017-145), the Netherlands. All participants will provide informed consent prior to participation in the study. Results of the study on primary and secondary outcome measures will be published in peer-reviewed journals. TRIAL REGISTRATION NUMBER: NTR6657; Pre-results.
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spelling pubmed-66157982019-07-28 A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial van den Heuvel, Lieke M Hoedemaekers, Yvonne M Baas, Annette F van Tintelen, J Peter Smets, Ellen M A Christiaans, Imke BMJ Open Genetics and Genomics INTRODUCTION: In current practice, probands are asked to inform relatives about the possibility of predictive DNA testing when a pathogenic variant causing an inherited cardiac condition (ICC) is identified. Previous research on the uptake of genetic counselling and predictive DNA testing in relatives suggests that not all relatives are sufficiently informed. We developed a randomised controlled trial to evaluate the effectiveness of a tailored approach in which probands decide together with the genetic counsellor which relatives they inform themselves and which relatives they prefer to have informed by the genetic counsellor. Here, we present the study protocol of this randomised controlled trial. METHODS: A multicentre randomised controlled trial with parallel-group design will be conducted in which an intervention group receiving the tailored approach will be compared with a control group receiving usual care. Adult probands diagnosed with an ICC in whom a likely pathogenic or pathogenic variant is identified will be randomly assigned to the intervention or control group (total sample: n=85 probands). Primary outcomes are uptake of genetic counselling and predictive DNA testing by relatives (total sample: n=340 relatives). Secondary outcomes are appreciation of the approach used and impact on familial and psychological functioning, which will be assessed using questionnaires. Relatives who attend genetic counselling will be asked to fill out a questionnaire as well. ETHICS AND DISSEMINATION: Ethical approval was obtained from the Medical Ethical Committee of the Amsterdam University Medical Centres (MEC 2017-145), the Netherlands. All participants will provide informed consent prior to participation in the study. Results of the study on primary and secondary outcome measures will be published in peer-reviewed journals. TRIAL REGISTRATION NUMBER: NTR6657; Pre-results. BMJ Publishing Group 2019-07-09 /pmc/articles/PMC6615798/ /pubmed/31289060 http://dx.doi.org/10.1136/bmjopen-2018-025660 Text en © Author(s) (or their employer(s)) 2019. Re-use permitted under CC BY. Published by BMJ. This is an open access article distributed in accordance with the Creative Commons Attribution 4.0 Unported (CC BY 4.0) license, which permits others to copy, redistribute, remix, transform and build upon this work for any purpose, provided the original work is properly cited, a link to the licence is given, and indication of whether changes were made. See: https://creativecommons.org/licenses/by/4.0/.
spellingShingle Genetics and Genomics
van den Heuvel, Lieke M
Hoedemaekers, Yvonne M
Baas, Annette F
van Tintelen, J Peter
Smets, Ellen M A
Christiaans, Imke
A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial
title A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial
title_full A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial
title_fullStr A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial
title_full_unstemmed A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial
title_short A tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial
title_sort tailored approach towards informing relatives at risk of inherited cardiac conditions: study protocol for a randomised controlled trial
topic Genetics and Genomics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6615798/
https://www.ncbi.nlm.nih.gov/pubmed/31289060
http://dx.doi.org/10.1136/bmjopen-2018-025660
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