The attitudes of people with sarcoma and their family towards genomics and incidental information arising from genetic research

Abstract

PurposeThe study aimed to examine attitudes of individuals diagnosed with sarcoma and their family members towards genetics, genomic research and incidental information arising as a result of participating in genetic research.MethodsA questionnaire was administered to 1200 individuals from the International Sarcoma Kindred Study (ISKS). Respondents were divided into three groups: individuals affected with sarcoma (probands), their spouses and family members.ResultsApproximately half of all research participants felt positively towards new discoveries in human genetics. Overall, more were positive in their attitudes towards genetic testing for inherited conditions (60%) but family members were less so. Older participants reported more highly positive attitudes more often than younger participants. Males were less likely to feel positive about new genetic discoveries and more likely to believe they could modify genetic risk by altering lifestyle factors. Almost all ISKS participants believed participants would like to be given ancillary information arising as a result of participating in genetic research.ConclusionsThe only difference between the study groups was the decreased likelihood of family members being highly positive about genetic testing. This may be important if predictive testing for sarcoma becomes available. Generally ISKS research participants supported the notion of returning incidental genetic information to research participants.

Attitudes of the general public towards the disclosure of individual research results and incidental findings from biobank genomic research in Australia

“…11 These findings are broadly consistent with a more recent and larger study amongst tissue donors arising from the International Sarcoma Kindred Study that examined attitudes towards genomic research amongst individuals diagnosed with sarcoma, their blood relatives and proband spouses. 20 This study found that all donors wanted to receive information (90-94%) about monogenic or polygenic conditions in which risk could be modified, but that for monogenic conditions that were not preventable, sarcoma probands and blood relatives were less likely to want to receive information than non-blood relative spouses (66.5% vs 76%, P = 0.05).…”

“…It is vital, therefore, that policy development in this area is democratic, evidence‐based and explicitly acknowledges the important ethical principles at stake, including autonomy, reciprocity and solidarity. Reassuringly, important research and policy reform are already being done in this area – with Australian oncology biobanks recently outlining several models and practical strategies for managing disclosure of IRR and IF which aim to preserve and respect the autonomy of biobank donors while at the same time acknowledging the necessity for scientific judgements to be made about the utility and importance of research findings in different clinical and research settings. Similar research and policy development continues internationally, including by the American College of Clinical Genetics and Genomics, the Association for Molecular Pathology the NIH funded International Sarcoma Kindred Study group, the Presidential Commission for the Study of Bioethical Issues and the UK Biobank.…”

“…It is vital, therefore, that policy development in this area is democratic, evidence-based and explicitly acknowledges the important ethical principles at stake, including autonomy, reciprocity and solidarity. Reassuringly, important research and policy reform are already being done in this areawith Australian oncology biobanks 20,25 recently outlining several models and practical strategies for managing disclosure of IRR and IF which aim to preserve and respect the autonomy of biobank donors while at the same time acknowledging the necessity for scientific judgements to be made about the utility and importance of research findings in different clinical and research settings. 1,26 More generally, however, this research provides clear evidence that knowledge gained in the course of research is valuable, in different ways to different stakeholders, and that advances in genomic research will be most likely to be successfully, and to be publicly supported, when the structures that support it are scientifically robust, clinically relevant and ethically sound.…”

Timing and context: important considerations in the return of genetic results to research participants

“…Studies suggest research participants are interested in receiving individual research results believing researchers have an obligation to return them (Bollinger et al 2012). Participants also want 'actionable' and accurate genetic research results returned (Murphy et al 2008;Young et al 2013). The term 'actionable' can have a different meaning to researchers and research participants.…”

“…Participants may have a different view and also regard personal utility as being part of actionability. This includes feeling a sense of control and helping to plan for the future (Young et al 2013;Kopits et al 2011).…”