Genetics in paediatric oncology is becoming increasingly important in diagnostics, treatment and follow-up care. Genetic testing may offer a possibility to stratify survivors follow-up care. However, survivors’ and parents’ preferences and needs for genetics-related services are largely unknown. This mixed-methods study assessed genetics-related information and service needs of survivors and parents. Six hundred and twenty-two participants (404 survivors: mean age: 26.27 years; 218 parents of survivors: mean age of child: 13.05 years) completed questionnaires. Eighty-seven participants (52 survivors; 35 parents) also completed in-depth telephone interviews. We analysed data using multivariable logistic regression and qualitative thematic analyses. Thirty-six of 50 families who were offered cancer-related genetic testing chose to undergo testing. Of those not offered testing, 11% of survivors and 7.6% of parents indicated that they believed it was ‘likely/very likely’ that the survivor had inherited a gene fault. Twenty-nine percent of survivors and 36% of parents endorsed access to a genetics specialist as important in their care. Survivors (40.9%) and parents (43.7%) indicated an unmet need for information about genetics and childhood cancer. Parents indicated a higher unmet need for information related to the survivors’ future offspring than survivors (p < 0.001). Many survivors and parents have unmet needs for genetics-related services and information. Greater access to services and information might allow survivors at high risk for late effects to detect and prevent side effects early and improve medical outcomes. Addressing families’ needs and preferences during survivorship may increase satisfaction with survivorship care.
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We thank Emma Doolan, Lucy Hanlon, Gabrielle Georgiou, Alison Young, Lachlan Munro, Luke Fry and Sanaa Mathur for their contributions to this research. We also thank each of the recruiting sites for this study, including Sydney Children’s Hospital Randwick, the Children’s Hospital at Westmead, John Hunter Children’s Hospital, the Royal Children’s Hospital Melbourne, Monash Children’s Hospital Melbourne, Lady Cilento Children’s Hospital Brisbane (formerly Royal Children’s Hospital Brisbane), Perth Children’s Hospital (formerly Princess Margaret Hospital for Children), Women’s and Children’s Hospital Adelaide, and in New Zealand, Starship Children’s Health, Wellington Hospital and Christchurch Hospital. We thank Joanna Fardell (PhD) and Kate Hetherington (PhD) for conducting some interviews. We thank Mark Donoghoe for his statistical support. CEW is supported by a Career Development Fellowship from the NHMRC of Australia (APP1143767). CS is supported by The Kids’ Cancer Project. The Behavioural Sciences Unit (BSU) is proudly supported by the Kids with Cancer Foundation.
*ANZCHOG Survivorship Study Group:
*The members of the ANZCHOG Survivorship Study Group in alphabetical order: Dr Frank Alvaro, Professor Richard Cohn, Dr Rob Corbett, Dr Peter Downie, Ms Karen Egan, Ms Sarah Ellis, Professor Jon Emery, Dr Joanna Fardell, Ms Tali Foreman, Dr Melissa Gabriel, Professor Afaf Girgis, Ms Kerrie Graham, Ms Karen Johnston, Dr Janelle Jones, Dr Liane Lockwood, Dr Ann Maguire, Dr Maria McCarthy, Dr Jordana McLoone, Dr Francoise Mechinaud, Ms Sinead Molloy, Ms Lyndal Moore, Dr Michael Osborn, Dr Christina Signorelli, Dr Jane Skeen, Dr Heather Tapp, Ms Tracy Till, Ms Jo Truscott, Ms Kate Turpin, Professor Claire Wakefield, Dr Thomas Walwyn, Ms Jane Williamson and Ms Kathy Yallop.
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Vetsch, J., Wakefield, C.E., Tucker, K.M. et al. Genetics-related service and information needs of childhood cancer survivors and parents: a mixed-methods study. Eur J Hum Genet (2019) doi:10.1038/s41431-019-0481-7