Journal article

"It's about having the choice": Stakeholder perceptions of population-based genetic carrier screening for fragile X syndrome

AD Archibald, CL Hickerton, AM Jaques, S Wake, J Cohen, SA Metcalfe

American Journal of Medical Genetics Part A | WILEY | Published : 2013

Abstract

This project explored, the views of key stakeholders regarding population-based genetic carrier screening for fragile X syndrome (FXS). Interviews and focus groups were conducted with healthcare providers, relatives of individuals with FXS and members of the general population. Data were transcribed verbatim and coded into themes. 188 individuals took part in this study. Perceived benefits of carrier screening included: learning the risk of having a child with FXS; learning the risk of fragile X-associated primary ovarian insufficiency; and the opportunity for carriers to access reproductive options. Concerns included: the emotional impact of screening and receiving a carrier result; the pre..

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Funding Acknowledgements

The authors would like to express their appreciation to the participants for taking the time to share their views and perspectives with us. We would like to thank Yasmin Bylstra for her assistance with data collection in the early stages of this project and A /Prof Allyn McConkie Rosell for her hellpful comments on an earlier version of this manuscript. We thank Victorian Clinical Genetics Services and the Fragile X Alliance Inc. for allowing us to recruit relatives of people with FXS through their services. This work was supported by the Fragile X Alliance Inc., Murdoch Childrens Research Institute and the Victorian Government's Operational Infrastructure Support Program. Dr Alison Archibald was supported by an Australian Postgraduate Award scholarship.