Supportive oncofertility care, psychological health and reproductive concerns: a qualitative study.

PURPOSE: Impaired fertility in cancer patients and survivors of reproductive age (15-45 years) may lead to psychological distress and poor mental health outcomes, and may negatively impact quality of life. Limited research has focused on the fertility experiences of those who have had access to supportive oncofertility care. This study aims to explore the fertility-care experiences and reproductive concerns of reproductive age cancer patients at the time of their cancer diagnosis who have had access to oncofertility care. METHODS: The qualitative data from a larger mixed method study is presented, comprising 30 semi-structured telephone interviews with newly diagnosed cancer patients across Australia and New Zealand, undertaken between April 2016 and April 2018. RESULTS: Interviews were undertaken with 9 male patients and 21 female patients aged between 15 and 44 years. All patients recalled a discussion about fertility and majority underwent some form of fertility preservation. Thematic analysis identified five main themes: (i) satisfaction with oncofertility care, (ii) a need for individualised treatment and support, (iii) desire for parenthood, (iv) fertility treatment can be challenging, and (v) fertility preservation provides a safety net for the future. CONCLUSIONS: Participants who access supportive oncofertility care report low emotional impact of threatened future infertility at the time of cancer diagnosis. These results suggest that such services may assist in lowering the emotional burden of potential infertility in survivors. Long-term research is needed to assess the longitudinal benefits for different models of care.

Children and young people's understanding of inherited conditions and their attitudes towards genetic testing: A systematic review.

Children and young people are increasingly likely to receive information regarding inherited health risks relevant to their genetic relatives and themselves. We reviewed the literature to determine what children and young people (21 years and younger) understand about inherited conditions and their attitudes towards genetic testing. We screened 1815 abstracts to identify 20 studies representing the perspectives of 1811 children and young people between the ages of 6 and 21 years (1498 children or young people at general population-level risk from 9 studies, 313 affected/at risk from 15 studies). Children and young people at general population-level risk demonstrated a basic understanding that disease predisposition can be inherited within families. Those affected by or at risk of genetic conditions inferred their genetic status from observable, relational characteristics within their family and the results of personal genetic testing if it had occurred, but some misunderstandings of important genetic concepts were evident. Children and young people expressed interest in and a willingness to undertake personal genetic testing, but also articulated concerns about the limitations and risks of testing. Paediatric patients require developmentally-sensitive genetic counselling and support in navigating the unique landscape of their condition.

'Why us?' Causal attributions of childhood cancer survivors, survivors' parents and community comparisons - a mixed methods analysis.

INTRODUCTION: Understanding the cause of their cancer is important for many cancer patients. Childhood cancer survivors'/survivors' parents' beliefs about cancer etiology are understudied. We aimed to assess survivors'/parents' beliefs about what causes childhood cancer, compared with beliefs in the community. We also investigated the influence of clinical and socio-demographic characteristics on the participants' beliefs about cancer etiology. METHODS: This two-stage study investigated the participants' beliefs, by using questionnaires assessing causal attributions related to childhood cancer (stage 1) and then undertaking telephone interviews (stage 2; survivors/survivors' parents only) to get an in-depth understanding of survivors'/survivors' parents beliefs. We computed multivariable regressions to identify factors associated with the most commonly endorsed attributions: bad luck/chance, environmental factors and genetics. We analyzed interviews using thematic analysis. RESULTS: Six hundred one individuals (64.6% survivors and 35.4% survivors' parents) and 510 community comparisons (53.1% community adults, 46.9% community parents) completed the question on causal attributions. We conducted 87 in-depth interviews. Survivors/survivors' parents (73.9%) were more likely to believe that chance/bad luck caused childhood cancer than community participants (42.4%). Community participants more frequently endorsed that genetics (75.3%) and environmental factors (65.3%) played a major role in childhood cancer etiology (versus survivors' and survivors' parents: genetics 20.6%, environmental factors: 19.3%). Community participants, participants with a first language other than English, and reporting a lower quality of life were less likely to attribute bad luck as a cause of childhood cancer. Community participants, all participants with a higher income and higher education were more likely to attribute childhood cancer etiology to environmental factors. CONCLUSION: Causal attributions differed between survivors/survivors' parents and community participants. Most of the parents and survivors seem to understand that there is nothing they have done to cause the cancer. Understanding survivors' and survivors' parents' causal attributions may be crucial to address misconceptions, offer access to services and to adapt current and future health behaviors.

Grandparents of children with cancer: a controlled comparison of perceived family functioning.

PURPOSE: Grandparents can be profoundly emotionally affected when a grandchild is diagnosed with cancer. They also often provide invaluable support for the family (e.g., caring for the sick child and/or siblings). Multigenerational family functioning may therefore change. Limited research has assessed grandparents' perspectives after their grandchild is diagnosed with cancer. In this study, we aimed to (1) assess differences in perceived family functioning among grandparents of a child with cancer and grandparents of healthy children and (2) assess the cancer-specific and demographic factors related to perceived family functioning in grandparents of a grandchild with cancer. PROCEDURE: Grandparents of a child with cancer (n = 89) and grandparents of healthy children (n = 133) completed the general functioning, communication, and problem-solving scales of the Family Assessment Device. We used multilevel models with a random intercept to detect (1) between-group differences and (2) identify factors related to perceived family functioning among grandparents with a grandchild with cancer. RESULTS: Grandparents with a grandchild with cancer reported poorer family functioning than grandparents with healthy grandchildren. Among the grandparents with a grandchild with cancer, impairments in family functioning were correlated with fewer years since diagnosis, providing care to their sick grandchild and/or siblings and living far away from the sick grandchild. CONCLUSIONS: The detrimental impact of childhood cancer likely extends beyond the immediate family members. Including grandparents in interventions-beginning at diagnosis-to reduce distress and increase cohesion for families of a child with cancer is warranted, particularly for grandparents who provide care to their sick grandchild or siblings.

Brief report: Bereaved parents informing research design: The place of a pilot study.

BACKGROUND: Risk minimization in research with bereaved parents is important. However, little is known about which research methods balance the sensitivity required for bereaved research participants and the need for generalizable results. AIM: To explore parental experiences of participating in mixed method bereavement research via a pilot study. DESIGN: A convergent parallel mixed method design assessing bereaved parents' experience of research participation. SETTING/PARTICIPANTS: Eleven parents whose child was treated for cancer at The Royal Children's Hospital, Brisbane completed the questionnaire/interview being piloted (n = 8 mothers; n = 3 fathers; >6 months and <6 years bereaved). Of these, eight parents completed the pilot study evaluation questionnaire, providing feedback on their experience of participation. RESULTS: Participants acknowledged the importance of bereaved parents being central to research design and the development of bereavement programs. Sixty-three per cent (n = 5/8) of parents described completion of the questionnaire as 'not at all/a little bit' of a burden. Seventy-five per cent (n = 6/8) of parents opting into the telephone interview described participation as 'not at all/a little bit' of a burden. When considering the latest timeframes for participation in bereavement research 63% (n = 5/8) of parents indicated 'no endpoint.' Findings from the pilot study enabled important adjustments to be made to a large-scale future study. CONCLUSIONS: As a research method, pilot studies may be utilized to minimize harm and maximize the potential benefits for vulnerable research participants. A mixed method approach allows researchers to generalize findings to a broader population while also drawing on the depth of the lived experience.

Health-related quality of life of survivors of childhood acute lymphoblastic leukemia: a systematic review.

PURPOSE: Acute lymphoblastic leukemia (ALL) survivors are the largest group of childhood cancer survivors; however, their risk for late effects is high. Cancer-related late effects have the potential to compromise health-related quality of life (HRQL) long into survivorship. None of the reviews so far have focused on ALL solely, but described HRQL for all childhood cancers. We aimed to identify ALL survivors at risk for poor HRQL and identify possible risk factors. METHOD: Following PRISMA guidelines, we performed a systematic review, searching published literature in Pubmed, PsycInfo, Embase, and the Cochrane database including all publications up to December 16, 2016. Two independent reviewers (JV and ER) screened eligible articles and assessed article quality. RESULTS: We found 31 studies representing 4356 survivors and 901 proxies. Thirteen studies found worse, eight found no difference, and three better, overall HRQL scores compared with healthy controls or norms. ALL survivors typically had better overall HRQL scores than survivors of other childhood cancers. Clinical variables (e.g., treatment received) were not consistently associated with HRQL; however, experiencing worse late effects was associated with lower HRQL. Survivor and parent socio-demographic factors and psychological factors such as resilience and depression were also associated with HRQL. CONCLUSION: ALL survivors appeared to have worse or equivalent HRQL compared with controls, but better HRQL than survivors of other cancer types. However, studies reported a wide variability in HRQL and potential risk factors for poor HRQL. Measuring ALL survivors' HRQL longitudinally and comprehensively assessing potential risk factors might identify future avenues to intervene early.

Parents' attitudes toward genetic testing of children for health conditions: A systematic review.

This review assessed parents' attitudes toward childhood genetic testing for health conditions, with a focus on perceived advantages and disadvantages. We also evaluated the factors that influence parents' attitudes toward childhood genetic testing. We searched Medline, Medline In-Process, EMBASE, PsycINFO, Social Work Abstracts and CINAHL. We screened 945 abstracts and identified 21 studies representing the views of 3934 parents. Parents reported largely positive attitudes toward childhood genetic testing across different genetic tests with varying medical utility. Parents perceived a range of advantages and disadvantages of childhood genetic testing. Childhood genetic testing was viewed by most as beneficial. Parents' education level, genetic status, sex and sociodemographic status were associated with reported attitudes. This yielded some conflicting findings, indicating the need for further research. Genetic counseling remains essential to support this population in making well-informed decisions. Targeted interventions tailored to specific families with different sociodemographic characteristics may be useful. Further research on the long-term impact of childhood genetic testing on families is warranted.

Grandparents of children with cancer: Quality of life, medication and hospitalizations.

BACKGROUND: Grandparents can play a crucial role of providing emotional and practical support for families facing childhood cancer. Yet, many have their own healthcare needs. This controlled study systematically assesses the impact of childhood cancer on grandparents' quality of life (QOL). Our objective was to compare QOL in grandparents of children with and without cancer and to identify factors associated with grandparents' QOL. PROCEDURE: Grandparents (N = 222) completed two patient-reported outcome (PRO) measures assessing QOL: EQ-5D-5L and WHOQOL-BREF. Secondary endpoints included sleep, medications and hospitalizations. We used independent samples t-tests and multivariate linear regression to assess between-group differences and identify predictors. RESULTS: Grandparents of children with cancer (n = 89) reported significantly worse QOL than controls (n = 133) [mean WHOQOL-BREF score: 75.6 (SD = 17.6) vs. 81.5 (15.6), P = 0.007; mean EQ-5D-5L index value: 0.777 (0.20) vs. 0.874 (0.14), P < 0.001)]. They also reported more problems with anxiety and depression (47.2 vs. 21.8%, P < 0.001) and pain (64.8 vs. 49.6%, P = 0.031). Grandparents of children with cancer reported taking longer to fall asleep [mean: 30.4 min (55.6) vs. 18.2 (20.2), P = 0.011] and taking more medications in the last 4 weeks [mean: 2.9 (SD = 3.8) vs. 1.8 (SD = 2.3), P = 0.012]. Hospitalizations were comparable across groups. Grandmothers, those living in urban locations, and retired/unemployed grandparents experienced reduced QOL. CONCLUSIONS: Grandparents are significantly affected by childhood cancer. The impact appears across many domains of life and results in meaningful QOL differences. Given that four or more individuals may be affected per child, and that grandparent well-being can influence the whole family, interventions targeting at-risk grandparents are needed.

Familial diagnostic experiences in paediatric oncology.

BACKGROUND: Diagnostic delays may not have significant prognostic implications in paediatric oncology, but psychological impacts remain understudied. METHODS: Interviews exploring diagnostic experiences were conducted with childhood cancer survivors (n=19), parents (n=78) and siblings (n=15). RESULTS: Median diagnostic time was 3 weeks. Participants described a mixture of rapid diagnoses (28.9%), plus delayed appraisal intervals (that is, parent- or patient-associated diagnostic delays; 40.0%) and diagnostic intervals (that is, healthcare-associated delays; 46.7%). Families experiencing delays described guilt and anger and deleterious impacts on the family-clinician relationship. Some believed delays impacted on treatment and prognosis. CONCLUSIONS: The effect of the diagnostic experience can be considerable.

Management of fever and neutropenia in children with cancer.

PURPOSE: Febrile neutropenia remains a common, life-threatening complication of chemotherapy in paediatric oncology. Delays in institution of empiric antibiotics have been identified at tertiary and regional centres caring for these patients and associated with decreased survival. Our objective was to reduce the time to administration of empiric antibiotics to less than 60 min from the time of presentation to hospital. METHODS: A retrospective study of the records of oncology patients presenting to the emergency department of a tertiary hospital over a 3-month period was performed and time to first antibiotic administration recorded. Potential causes of delay in commencement of antibiotics were identified and an algorithm-based approach to the management of fever in immunocompromised children developed and implemented. Follow-up evaluation data were collected at 12 and 60 months post-intervention. Causes of delay in commencement of antibiotics at regional hospitals that share care with the tertiary hospital were identified through questionnaires, interviews and focus groups, involving patients and medical and nursing staff. The impact of the introduction of the algorithm at one peripheral hospital was evaluated. RESULTS: The mean time to empiric antibiotics was reduced from 148 min (95% confidence interval (CI) 81-216) at baseline to 76 min (95% CI 50-101) at 12 months post-intervention and sustained at 65 min (95% CI 52-77) 5 years after the intervention. At the peripheral hospital, mean time to antibiotic delivery was reduced from 221 min (95% CI 114-328) to 65 min (95% CI 42-87) at 12 months after the intervention. CONCLUSION: The introduction of the guideline, with teaching and support for staff and parents, resulted in an improvement in practice, meeting international guidelines and achieving sustained results at 5 years after introduction at a tertiary hospital. The guideline has been shown to be feasible and effective at a regional hospital.

Psychometric evaluation of the Sibling Cancer Needs Instrument (SCNI): an instrument to assess the psychosocial unmet needs of young people who are siblings of cancer patients.

PURPOSE: The current study sought to establish the psychometric properties of the revised Sibling Cancer Needs Instrument (SCNI) when completed by young people who have a brother or sister with cancer. METHODS: The participants were 106 young people aged between 12 and 24 who had a living brother or sister diagnosed with any type or stage of cancer in the last 5 years. They were recruited from multiple settings. The initial step in determining the dimensional structure of the questionnaire was exploratory factor analysis and further assessment followed using Rasch analysis. Construct validity and test-retest reliability (n = 17) were also assessed. RESULTS: The final SCNI has 45 items and seven domains: information; practical assistance; "time out" and recreation; feelings; support (friends and other young people); understanding from my family; and sibling relationship. There was a reasonable spread of responses across the scale for every item. Rasch analysis results suggested that overall, respondents used the scale consistently. Support for construct validity was provided by the correlations between psychological distress and the SCNI domains. The internal consistency was good to excellent; Cronbach's alphas ranged from 0.78 to 0.94. The test-retest reliability of the overall measure is 0.88. CONCLUSIONS: The SCNI is the first measure of psychosocial unmet needs which has been developed for young people who have a brother or sister with cancer. The sound psychometric properties allow the instrument to be used with confidence. The measure will provide a substantial clinical benefit in highlighting the unmet needs of this population to assist with the prioritisation of targeted supportive care services and evaluating the impact of interventions targeted at siblings.

Childhood cancer survivors' school (re)entry: Australian parents' perceptions.

Starting or returning to school after intense medical treatment can be academically and socially challenging for childhood cancer survivors. This study aimed to evaluate the school (re)entry experience of children who had recently completed cancer treatment. Forty-two semi-structured telephone interviews were conducted to explore parents' perceptions of their child's (re)entry to school after completing treatment (23 mothers, 19 fathers, parent mean age 39.5 years; child mean age 7.76 years). Interviews were analysed using the framework of Miles and Huberman and emergent themes were organised using QSR NVivo8. Parents closely monitored their child's school (re)entry and fostered close relationships with their child's teacher to ensure swift communication of concerns should they arise. The most commonly reported difficulty related to aspects of peer socialisation; survivors either displayed a limited understanding of social rules such as turn taking, or related more to older children or teachers relative to their peers. Additionally, parents placed a strong emphasis on their child's overall personal development, above academic achievement alone. Improved parent, clinician and teacher awareness of the importance of continued peer socialisation during the treatment period is recommended in order to limit the ongoing ramifications this may have on school (re)entry post-treatment completion.

Support after the completion of cancer treatment: perspectives of Australian adolescents and their families.

Young people recovering from cancer may lack adequate support post-treatment, yet little is known about the types of support and information young Australians and their families need. This study investigated adolescent/young adult cancer survivors' and their families' perceptions of care and support needs after completing cancer treatment. Seventy semi-structured interviews were conducted with 19 survivors (mean age 16.1 years), 21 mothers, 15 fathers and 15 siblings. Interviews were recorded, transcribed and analysed using the conceptual framework of Miles and Huberman. Post-treatment, participants regarded medical staff positively but were reluctant to ask for their help fearing it may deflect resources away from patients still receiving treatment. Appraisals of social workers' and psychologists' support post-treatment were mixed. Formal emotional support was rarely accessed and participants reported that any additional funds should be directed to greater psychological support in this period. Participants also reported the need for additional financial support post-treatment. Clinicians need to be aware that while young people and their families may not demand support post-treatment, they may 'suffer in silence' or burden family members and friends with the responsibility of providing emotional support, though they may be experiencing distress also.

Hospitalizations in Australian children with neuroblastoma: A population-based study.

BACKGROUND: An increasing number of children diagnosed with both low- and high-risk neuroblastoma are surviving. Yet, treatment can be intensive and often multimodal, especially for high-risk neuroblastoma, resulting in significant long-term health problems. We aimed to describe neuroblastoma survivors' pediatric hospitalizations, readmissions, and their associated costs. METHOD: We conducted a population-based study of all children (<18 years) residing in New South Wales (NSW), Australia, and hospitalized with a recorded diagnosis of neuroblastoma during 2001-2020. We used linked NSW Admitted Patient Data Collection and death registration data to examine the frequency, length of stay, and readmissions following the first admission when neuroblastoma was diagnosed (i.e., the index admission), and the associated hospitalization costs by age and timing postindex admission discharge. RESULTS: In total, 300 children (64% aged <3 years) were hospitalized for neuroblastoma over the study period. The median number of readmissions and length of stay within 2 years postdischarge were 17 (interquartile range IQR: 5.5-25) and 45.5 (IQR: 10-125) days, and median cost per child was AUD$124,058 (IQR $34,217-$264,627). Following discharge from the index admission, there were 7088 readmissions (median: 20 per child, IQR: 7-29). Fifty-eight percent of readmissions occurred within 1-year postdischarge, primarily due to fever, nausea, abdominal pain, and respiratory conditions. CONCLUSION: The burden of health problems requiring hospitalization among neuroblastoma survivors results in significant associated healthcare costs, warranting further efforts to optimize health care for neuroblastoma survivors that focuses on early intervention and long-term monitoring.

Growth and Nutritional Outcomes in Children Post-Haematopoietic Stem Cell Transplant without Exposure to Total Body Irradiation.

AIMS: Poor growth in childhood cancer survivors who undergo haematopoietic stem cell transplant (HSCT) without exposure to radiation is reported anecdotally, although literature to support this is limited. The aims of this study were to assess the change in height standard deviation score (SDS) and the final adult height (FAH) in children who underwent chemotherapy-only conditioned HSCT and to identify predictors of poor growth. MATERIALS AND METHODS: We conducted a retrospective hospital medical record review (1984-2010) of children (1-10 years) who underwent chemotherapy-only conditioned HSCT, noting anthropology measurements at cancer diagnosis, HSCT, 10 years old and FAH. RESULTS: The median age at HSCT of the 53 patients was 4.5 years, 75% had a haematological malignancy and 25% a solid tumour. Half of the cohort underwent allogenic HSCT and most (89%) conditioned with busulphan. The mean change in height SDS from primary cancer diagnosis to FAH was -1.21 (±1.18 SD), equivalent to 7-8.5 cm loss, with a mean FAH of -0.91 SDS (±1.10 SD). The greatest height loss occurred between diagnosis and HSCT (-0.77 SDS, 95% confidence interval -1.42, -0.12, P = 0.01), with no catch-up growth seen by FAH. Patients with solid tumours had the greatest height loss. Overall body mass index SDS did not change significantly over time, or by cancer type. CONCLUSIONS: Chemotherapy-only conditioned HSCT during childhood can impact FAH, with the greatest height loss occurring prior to HSCT and no catch-up growth after treatment finishes. Children transplanted for a solid tumour malignancy seem to be more at risk, possibly due to intensive treatment regimens, both pre-transplant and during conditioning.

Reduced incidence of second solid tumors in survivors of childhood Hodgkin's lymphoma treated without radiation therapy.

BACKGROUND: We assessed the risk of developing second malignancies in children treated for Hodgkin's lymphoma (HL), the majority of whom received chemotherapy only. PATIENTS AND METHODS: The development of second malignancies in children with HL, treated between 1960 and 1999, was assessed. Results were obtained by both chart review and linkage with a centralized cancer registry. Tumor incidence was compared for patients treated with and without radiotherapy (RT) and with the general population. Risk factors for developing second tumors were assessed by multivariate analysis. RESULTS: Of 142 childhood HL patients, 63 had received RT and 79 had not. Overall survival was similar for both groups. Fourteen patients developed second solid tumors, 12 who had received RT and 2 treated with chemotherapy only (P <0.001), with a 30-year cumulative incidence of 24.7% [95% confidence interval (CI) 7.27-47.4] and 5.8% (95% CI 0-58.9), respectively (P = 0.01). The standardized incidence ratio for second solid tumors was 236 (95% CI 112.2-359.0) versus 43.6 (95% CI 0-103.9), respectively. Multivariate analysis showed treatment with RT was the only significant risks factor for developing second solid tumors. CONCLUSIONS: Children with HL without RT have a substantially lower incidence of second tumors than those treated with RT.

Changes in long term peripheral nerve biophysical properties in childhood cancer survivors following neurotoxic chemotherapy.

OBJECTIVE: In the context of increasing numbers of childhood cancer survivors (CCS), this study aimed to enhance understanding of the biophysical basis for long term chemotherapy induced peripheral neuropathy from different chemotherapy agents in CCS. METHODS: Detailed cross-sectional neurophysiological examination, using median nerve axonal excitability studies, alongside clinical assessments, in 103 long term CCS (10.5 ± 0.6 years post-treatment). RESULTS: Cisplatin treated CCS (n = 16) demonstrated multiple sensory axonal excitability changes including increased threshold (P < 0.05), alterations in depolarising and hyperpolarising threshold electrotonus (P < 0.05) and reduction in resting and minimum IV slope (P < 0.01). Vincristine treated CCS (n = 73) were comparable to controls, except for prolonged distal motor latency (P = 0.001). No differences were seen in the non-neurotoxic chemotherapy group (n = 14). Abnormalities were more evident in the cisplatin subgroup with greater clinical neuropathy manifestations. CONCLUSION: Persistent long term changes in axonal biophysical properties vary with different chemotherapy agents, most evident after cisplatin exposure. Longitudinal studies of nerve function during chemotherapy treatment are required to further evaluate these differences and their mechanistic basis. SIGNIFICANCE: This study provides a unique biophysical perspective for persistent cisplatin related neurotoxicity in children, previously under recognised.

Perceived cancer-related pain and fatigue, information needs, and fear of cancer recurrence among adult survivors of childhood cancer.

OBJECTIVE: Pain and fatigue are under-researched late effects of childhood cancer and its treatment, and may be interpreted by survivors as indicating cancer recurrence. Moreover, unmet information needs for managing pain and fatigue may be related to fear of cancer recurrence. We investigated the complex relationships between perceived cancer-related pain and fatigue, unmet information needs for managing pain and fatigue, and fear of cancer recurrence. METHODS: We surveyed 404 adult survivors of any form of childhood cancer (M = 16.82 years since treatment completion). RESULTS: Many survivors reported perceived cancer-related pain (28.7%) and fatigue (40.3%), and anticipated future pain (19.3%) and fatigue (26.2%). These symptomologies were all related to unmet information needs for managing pain (18.8%) and fatigue (32.2%; all p's<.001). Survivors reporting unmet information needs for managing pain (B = .48, 95% CI = 0.19-0.76, p = .001) and fatigue (B = .32, 95% CI = 0.06-0.52, p = .015) reported higher fear of cancer recurrence than survivors reporting no information needs. CONCLUSION: Survivors often have unmet information needs for managing pain and fatigue, and these unmet needs are related to fear of cancer recurrence. PRACTICE IMPLICATIONS: Long-term follow-up clinics should assess pain and fatigue. Information provision about pain and fatigue may be an important tool to help manage fear of cancer recurrence.

The Avatar Acceptability Study: Survivor, Parent and Community Willingness to Use Patient-Derived Xenografts to Personalize Cancer Care.

BACKGROUND: Using patient-derived xenografts (PDXs) to assess chemosensitivity to anti-cancer agents in real-time may improve cancer care by enabling individualized clinical decision-making. However, it is unknown whether this new approach will be met with acceptance by patients, family and community. METHODS: We used a cross-sectional structured survey to investigate PDX acceptability with 1550 individuals across Australia and New Zealand (648 survivors of adult and childhood cancer, versus 650 community comparisons; and 48 parents of childhood cancer survivors versus 204 community parents). We identified factors influencing willingness-to-use PDXs, willingness-to-pay, maximum acceptable wait-time, and maximum acceptable number of mice used per patient. FINDINGS: PDXs were highly acceptable: >80% of those affected by cancer felt the potential advantages of PDXs outweighed the disadvantages (community participants: 68%). Survivors' and survivors' parents' most highly endorsed advantage was 'increased chance of survival'. 'Harm to animals' was the least endorsed disadvantage for all groups. Cancer survivors were more willing to use PDXs than community comparisons [p < ·001]. Survivors and survivors' parents were willing to pay more [p < ·001; p = ∙004 respectively], wait longer for results [p = ·03; p = ∙01], and use more mice [p = ·01; p < ∙001] than community comparisons. Male survivors found PDXs more acceptable [p = ·01] and were willing to pay more [p < ·001] than female survivors. Survivors with higher incomes found PDXs more acceptable [p = ·002] and were willing to pay more [p < ·001] than survivors with lower incomes. Mothers found PDXs more acceptable [p = ·04] but were less willing to wait [p = ·02] than fathers. INTERPRETATION: We found significant attitudinal support for PDX-guided cancer care. Willingness-to-pay and maximum acceptable number of mice align well with likely future usage. Maximum acceptable wait-times were lower than is currently achievable, highlighting an important area for future patient education until technology has caught up.