Abstract Advances in our understanding of the genetic basis of childhood cancer, including Primary Central Nervous System (CNS) cancers, are improving the diagnosis, treatment and clinical management of pediatric patients. To effectively translate scientific breakthroughs into enhanced clinical care, it is essential we understand and learn from the experiences of patients, families, and health professionals. This report summarizes findings from four Australian psychosocial sub-studies exploring the perspectives of patients, parents, clinicians and scientists participating in research related to childhood cancer genetics. Specifically, these studies focus on the psychosocial impact of germline testing in children, surveillance for children with a cancer predisposition syndrome and the perspectives of healthcare professionals who deliver this testing and surveillance. Data presented highlight some of the opportunities and challenges associated with the changing context of genetic predisposition testing for children and AYAs with cancer and illustrate how embedding psychosocial data collection in clinical research can answer important questions in the field and inform the design of patient-centric models of care, resources, and workforce training. By embracing these perspectives, we can ensure that advances in genetic research translate into enhanced family experiences, and, ultimately, improved outcomes for children and young people with cancer, and their families.