Cargando…
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...
Autores principales: | , , , , , |
---|---|
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 |
_version_ | 1783433402565787648 |
---|---|
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. |
format | Online Article Text |
id | pubmed-6615798 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2019 |
publisher | BMJ Publishing Group |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT vandenheuvelliekem atailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT hoedemaekersyvonnem atailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT baasannettef atailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT vantintelenjpeter atailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT smetsellenma atailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT christiaansimke atailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT vandenheuvelliekem tailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT hoedemaekersyvonnem tailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT baasannettef tailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT vantintelenjpeter tailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT smetsellenma tailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial AT christiaansimke tailoredapproachtowardsinformingrelativesatriskofinheritedcardiacconditionsstudyprotocolforarandomisedcontrolledtrial |