Causation in cerebral palsy: Parental beliefs and associated emotions.

AIM: To better understand parents' beliefs about causation in cerebral palsy (CP) and the emotions related to those beliefs. METHOD: We surveyed 226 parents of children with CP aged 1 to 18 years, recruited from the Victorian Cerebral Palsy Register, to evaluate their beliefs about the causes of CP, including genetic causes, causes specific to their own child, and their attitudes and emotions in relation to these. RESULTS: Although 92% of participants reported that understanding the causes of their child's CP was important, uncertainty about the cause was expressed by 13%. The most frequently endorsed causal factors, in general and in their own child respectively, were intrapartum hypoxia (81%, 36%) or brain damage (69%, 22%), brain damage during pregnancy (73%, 28%), and preterm birth (66%, 28%). Genetic causes were deemed relevant by 13% of participants and hospital or professional error by 16%. Parents shared related feelings of anger (59%), sadness (80%), guilt (61%), and confusion (53%); parental anger was more likely when their child's CP was attributed to intrapartum events. INTERPRETATION: Substantial parental interest in understanding the causes of CP, together with uncertainty about the causes, parents' causal attributions, and significant emotional sequelae, highlight a strong need for provision of information and support for families of children recently diagnosed with CP. WHAT THIS PAPER ADDS: Understanding the causes of their child's cerebral palsy (CP) was important to parents. Parents most often endorsed intrapartum factors as a cause of CP. Parents reported experiencing strong emotions about the causes of their child's CP.

Surgeon reported measures of stress and anxiety prior to and after elective gynecological surgery.

INTRODUCTION: Performing surgical procedures is a recognized source of stress for surgeons. Vocational stress is an important contributor to performance, patient care, and burnout with dispositional and environmental factors contributing. Accurately assessing surgeon stress is critical to measuring effectiveness of stress reduction programs. The primary aim was to identify differences between surgeons' self-reported anticipated stress and anxiety prior to gynecological surgery, compared with their recollection of experienced stress and anxiety during surgery. Secondary aims assessed any differences by level of training, surgical type, and surgeon role. MATERIAL AND METHODS: Attending and resident gynecologists performing routine elective surgeries completed a visual analog scale (VAS) assessing perceived stress and the State-Trait Anxiety Inventory (STAI) prior to and immediately after completing 161 elective surgeries including total laparoscopic hysterectomy, laparoscopic excision of moderate-severe endometriosis, or hysteroscopic myomectomy. RESULTS: Eight attending gynecologists and nine residents participated. Residents commenced as primary surgeon in 62/90 (69%) procedures. Stress experienced during surgery was greater than anticipated in 92/161 (57%) surgery episodes (mean VAS increase: 3.9; 95% CI: 1.1-6.8, p = 0.009). State anxiety was greater than anticipated in 99/161 (62%) episodes (mean state anxiety increase: 4.4; 95% CI: 3.0-5.8, p < 0.001). Greater preprocedural anticipatory stress and anxiety was observed in residents vs. attending gynecologists (VAS 51.9 vs. 22.8, p < 0.001; state anxiety 38.3 vs. 28.1, p < 0.001) and in primary vs. assistant surgeons (VAS 47.2 vs. 29.9, p < 0.001; state anxiety 36.9 vs. 28.3, p < 0.001). Intraoperative stress and anxiety were greater in primary surgeons (VAS 50.4 vs. 30.5, p < 0.001; anxiety 41.3 vs. 32.5, p < 0.001) and residents (VAS 43.4 vs. 31.7, p < 0.001; anxiety 53.5 vs. 33.7, p < 0.001) compared with assistants and attending gynecologists. Perceived stress and anxiety were positively correlated at both timepoints (r = 0.68, p < 0.001; r = 0.82, p < 0.001). CONCLUSIONS: When asked to reflect on stress experienced during surgery, our data show that stress during surgery is greater than anticipated for many surgical episodes. Self-reported stress symptoms commence prior to surgery and are more commonly reported by surgeons operating as primary surgeon and by those in training. Future research should focus on determinants of presurgical stress and examine when stressors become inhibitory to performance.

'High hopes for treatment': Australian stakeholder perspectives of the clinical translation of advanced neurotherapeutics for rare neurological diseases.

INTRODUCTION: Advanced therapies offer unprecedented opportunities for treating rare neurological disorders (RNDs) in children. However, health literacy, perceptions and understanding of novel therapies need elucidation across the RND community. This study explored healthcare professionals' and carers' perspectives of advanced therapies in childhood-onset RNDs. METHODS: In this mixed-methodology cross-sectional study, 20 healthcare professionals (clinicians, genetic counsellors and scientists) and 20 carers completed qualitative semistructured interviews and custom-designed surveys. Carers undertook validated psychosocial questionnaires. Thematic and quantitative data analysis followed. RESULTS: Participants described high positive interest in advanced therapies, but low knowledge of, and access to, reliable information. The substantial 'therapeutic gap' and 'therapeutic odyssey' common to RNDs were recognised in five key themes: (i) unmet need and urgency for access; (ii) seeking information; (iii) access, equity and sustainability; (iv) a multidisciplinary and integrated approach to care and support and (v) difficult decision-making. Participants were motivated to intensify RND clinical trial activity and access to advanced therapies; however, concerns around informed consent, first-in-human trials and clinical trial procedures were evident. There was high-risk tolerance despite substantial uncertainties and knowledge gaps. RNDs with high mortality, increased functional burdens and no alternative therapies were consistently prioritised for the development of advanced therapies. However, little consensus existed on prioritisation to treatment access. CONCLUSIONS: This study highlights the need to increase clinician and health system readiness for the clinical translation of advanced therapeutics for RNDs. Co-development and use of educational and psychosocial resources to support clinical decision-making, set therapeutic expectations and promotion of equitable, effective and safe delivery of advanced therapies are essential. PATIENT OR PUBLIC CONTRIBUTION: Participant insights into the psychosocial burden and information need to enhance the delivery of care in this formative study are informing ongoing partnerships with families, including co-production and dissemination of psychoeducational resources featuring their voices hosted on the Sydney Children's Hospitals Network website SCHN Brain-Aid Resources.

Neuroblastoma survivors' self-reported late effects, quality of life, health-care use, and risk perceptions.

BACKGROUND: Survivors of childhood neuroblastoma are at risk of multiple treatment-related health problems (late effects), impacting their quality of life. While late effects and quality of life among Australia and New Zealand (ANZ) childhood cancer survivors have been reported, the outcomes of neuroblastoma survivors specifically have not been reported, limiting critical information to inform treatment and care. METHODS: Young neuroblastoma survivors or their parents (as proxy for survivors <16 years) were invited to complete a survey and optional telephone interview. Survivors' late effects, risk perceptions, health-care use, and health-related quality of life were surveyed and analyzed using descriptive statistics and linear regression analyses. In-depth interviews explored participants' experiences, knowledge, and perception of late effects and information needs. Thematic content analysis was used to summarize the data. RESULTS: Thirty-nine neuroblastoma survivors or parents completed questionnaires (median age = 16 years, 39% male), with 13 also completing interviews. Thirty-two participants (82%) reported experiencing at least 1 late effect, most commonly dental problems (56%), vision/hearing problems (47%), and fatigue (44%). Participants reported high overall quality of life (index = 0.9, range = 0.2-1.0); however, more participants experienced anxiety/depression compared to the population norm (50% met criteria versus 25%, χ2 = 13, p < 0.001). Approximately half of participants (53%) believed they were at risk of developing further late effects. Qualitatively, participants reported knowledge gaps in understanding their risk of developing late effects. CONCLUSION: Many neuroblastoma survivors appear to experience late effects, anxiety/depression and have unmet cancer-related information needs. This study highlights important areas for intervention to reduce the impact of neuroblastoma and its treatment in childhood and young adulthood.

Communicating with families of young people with hard-to-treat cancers: Healthcare professionals' perspectives on challenges, skills, and training.

OBJECTIVES: Hard-to-treat childhood cancers are those where standard treatment options do not exist and the prognosis is poor. Healthcare professionals (HCPs) are responsible for communicating with families about prognosis and complex experimental treatments. We aimed to identify HCPs' key challenges and skills required when communicating with families about hard-to-treat cancers and their perceptions of communication-related training. METHODS: We interviewed Australian HCPs who had direct responsibilities in managing children/adolescents with hard-to-treat cancer within the past 24 months. Interviews were analyzed using qualitative content analysis. RESULTS: We interviewed 10 oncologists, 7 nurses, and 3 social workers. HCPs identified several challenges for communication with families including: balancing information provision while maintaining realistic hope; managing their own uncertainty; and nurses and social workers being underutilized during conversations with families, despite widespread preferences for multidisciplinary teamwork. HCPs perceived that making themselves available to families, empowering them to ask questions, and repeating information helped to establish and maintain trusting relationships with families. Half the HCPs reported receiving no formal training for communicating prognosis and treatment options with families of children with hard-to-treat cancers. Nurses, social workers, and less experienced oncologists supported the development of communication training resources, more so than more experienced oncologists. SIGNIFICANCE OF RESULTS: Resources are needed which support HCPs to communicate with families of children with hard-to-treat cancers. Such resources may be particularly beneficial for junior oncologists and other HCPs during their training, and they should aim to prepare them for common challenges and foster greater multidisciplinary collaboration.

Australian Headache Epidemiology Data (AHEAD): a pilot study to assess sampling and engagement methodology for a nationwide population-based survey.

BACKGROUND: There are no robust population-based Australian data on prevalence and attributed burden of migraine and medication-overuse headache (MOH) data. In this pilot cross-sectional study, we aimed to capture the participation rate, preferred response method, and acceptability of self-report questionnaires to inform the conduct of a future nationwide migraine/MOH epidemiological study. METHODS: We developed a self-report questionnaire, available in hard-copy and online, including modules from the Headache-Attributed Restriction, Disability, Social Handicap and Impaired Participation (HARDSHIP) questionnaire, the Eq. 5D (quality of life), and enquiry into treatment gaps. Study invitations were mailed to 20,000 randomly selected households across Australia's two most populous states. The household member who most recently had a birthday and was aged ≥ 18 years was invited to participate, and could do so by returning a hard-copy questionnaire via reply-paid mail, or by entering responses directly into an online platform. RESULTS: The participation rate was 5.0% (N = 1,000). Participants' median age was 60 years (IQR 44-71 years), and 64.7% (n = 647) were female. Significantly more responses were received from areas with relatively older populations and middle-level socioeconomic status. Hard copy was the more commonly chosen response method (n = 736). Females and younger respondents were significantly more likely to respond online than via hard-copy. CONCLUSIONS: This pilot study indicates that alternative methodology is needed to achieve satisfactory engagement in a future nationwide migraine/MOH epidemiological study, for example through inclusion of migraine screening questions in well-resourced, interview-based national health surveys that are conducted regularly by government agencies. Meanwhile, additional future research directions include defining and addressing treatment gaps to improve migraine awareness, and minimise under-diagnosis and under-treatment.

Relationship between survivorship care plans and unmet information needs, quality of life, satisfaction with care, and propensity to engage with, and attend, follow-up care.

BACKGROUND: The impact of survivorship care plans (SCPs) on the proximal and distal outcomes of adult and childhood cancer survivors, and parent proxies, is unclear. This study aimed to determine the relationship between SCP receipt and these outcomes. METHODS: A cross-sectional survey of adult and childhood cancer survivors (and parent proxies for survivors aged younger than 16 years) across Australia and New Zealand was conducted. Multivariate regression models were fitted to measure the impact of SCP receipt on proximal (unmet information needs and propensity to engage with, and attend, cancer-related follow-up care) and distal outcomes (quality of life and satisfaction with cancer-related follow-up care) with control for cancer history and sociodemographic factors. RESULTS: Of 1123 respondents, 499 were adult cancer survivors and 624 were childhood cancer survivors (including 222 parent proxies). We found that SCP receipt was predictive of greater attendance at, and awareness of, cancer-related follow-up care (adult: odds ratio [OR], 2.46; 95% CI, 1.18-5.12; OR, 2.38; 95% CI, 1.07-5.29; child/parent: OR, 2.61; 95% CI, 1.63-4.17; OR, 1.63; 95% CI, 1.06-2.50; respectively). SCP receipt also predicted fewer unmet information needs related to "follow-up care required" and "possible late effects" (adult: OR, 0.44; 95% CI, 0.20-0.96; OR, 0.29; 95% CI, 0.13-0.64; child/parent: OR, 0.46; 95% CI, 0.30-0.72; OR, 0.57; 95% CI, 0.38-0.85; respectively). In terms of distal outcomes, SCP receipt predicted a better global quality of life for adult cancer survivors (ß, 0.08; 95% CI, -0.01-7.93), proxy-reported health-related quality of life (ß, 0.15; 95% CI, 0.44-7.12), and satisfaction with follow-up care for childhood cancer survivors (OR, 2.93; 95% CI, 1.64-5.23). CONCLUSIONS: Previous studies have shown little impact of SCPs on distal end points. Results suggest that SCPs may be beneficial to cancer survivors' proximal and distal outcomes.

Parents' and adolescents' perspectives and understanding of information about childhood cancer precision medicine.

BACKGROUND: Precision medicine is projected to become integral to childhood cancer care. As such, it is essential to support families to understand what precision medicine entails. METHODS: A total of 182 parents and 23 adolescent patients participating in Precision Medicine for Children with Cancer (PRISM), an Australian precision medicine clinical trial for high-risk childhood cancer, completed questionnaires after study enrollment (time 0 [T0]). Of the parents, 108 completed a questionnaire and 45 completed an interview following return of precision medicine results (time 1 [T1]). We analyzed the mixed-methods data comprising measures exploring families' perceptions and understanding of PRISM's participant information sheet and consent form (PISCF), and factors associated with understanding. RESULTS: Most parents were satisfied with the PISCF, rating it as at least "somewhat" clearly presented (n = 160/175; 91%) and informative (n = 158/175; 90%). Many suggested improvements including the use of clearer language and a more visually engaging format. Parents' actual understanding of precision medicine was low on average, but scores improved between T0 and T1 (55.8/100-60.0/100; p = .012). Parents from culturally and/or linguistically diverse backgrounds (n = 42/177; 25%) had lower actual understanding scores than those from a Western/European background whose first language was English (p = .010). There was little correlation between parents' perceived and actual understanding scores (p = .794; Pearson correlation -0.020; 95% CI, -0.169 to 0.116). Most adolescent patients read the PISCF either "briefly" or "not at all" (70%) and had a perceived understanding score of 63.6/100 on average. CONCLUSIONS: Our study revealed gaps in families' understanding of childhood cancer precision medicine. We highlighted areas for potential intervention such as through targeted information resources. PLAIN LANGUAGE SUMMARY: Precision medicine is projected to become part of the standard of care for children with cancer. Precision medicine aims to give the right treatment to the right patient and involves several complex techniques, many of which may be challenging to understand. Our study analyzed questionnaire and interview data from parents and adolescent patients enrolled in an Australian precision medicine trial. Findings revealed gaps in families' understanding of childhood cancer precision medicine. Drawing on parents' suggestions and the literature, we make brief recommendations about improving information provision to families, such as through targeted information resources.

Parents' expectations, preferences, and recall of germline findings in a childhood cancer precision medicine trial.

BACKGROUND: Germline genome sequencing in childhood cancer precision medicine trials may reveal pathogenic or likely pathogenic variants in cancer predisposition genes in more than 10% of children. These findings can have implications for diagnosis, treatment, and the child's and family's future cancer risk. Understanding parents' perspectives of germline genome sequencing is critical to successful clinical implementation. METHODS: A total of 182 parents of 144 children (<18 years of age) with poor-prognosis cancers enrolled in the Precision Medicine for Children with Cancer trial completed a questionnaire at enrollment and after the return of their child's results, including clinically relevant germline findings (received by 13% of parents). Parents' expectations of germline genome sequencing, return of results preferences, and recall of results received were assessed. Forty-five parents (of 43 children) were interviewed in depth. RESULTS: At trial enrollment, most parents (63%) believed it was at least "somewhat likely" that their child would receive a clinically relevant germline finding. Almost all expressed a preference to receive a broad range of germline genomic findings, including variants of uncertain significance (88%). Some (29%) inaccurately recalled receiving a clinically relevant germline finding. Qualitatively, parents expressed confusion and uncertainty after the return of their child's genome sequencing results by their child's clinician. CONCLUSIONS: Many parents of children with poor-prognosis childhood cancer enrolled in a precision medicine trial expect their child may have an underlying cancer predisposition syndrome. They wish to receive a wide scope of information from germline genome sequencing but may feel confused by the reporting of trial results.

Hopes, concerns, satisfaction and regret in a precision medicine trial for childhood cancer: a mixed-methods study of parent and patient perspectives.

BACKGROUND: Paediatric precision oncology aims to match therapeutic agents to driver gene targets. We investigated whether parents and patients regret participation in precision medicine trials, particularly when their hopes are unfulfilled. METHODS: Parents and adolescent patients completed questionnaires at trial enrolment (T0) and after receiving results (T1). Parents opted-in to an interview at T1. Bereaved parents completed a questionnaire 6-months post-bereavement (T1B). We analysed quantitative data with R and qualitative data thematically with NVivo, before integrating all data for interpretation. RESULTS: 182 parents and 23 patients completed T0; 108/182 parents and 8/23 patients completed T1; 27/98 bereaved parents completed T1B; and 45/108 parents were interviewed. At enrolment, participants held concurrent hopes that precision medicine would benefit future children and their child. Participants expressed concern regarding wait-times for receipt of results. Most participants found the trial beneficial and not burdensome, including bereaved parents. Participants reported high trial satisfaction (median scores: parents: 93/100; patients: 80/100). Participants expressed few regrets (parent median scores: parents: 10/100; bereaved parents: 15/100; patient regret: 2/8 expressed minimal regret). CONCLUSIONS: Even when trial outcomes did not match their hopes, parents and patients rarely regretted participating in a childhood cancer precision medicine trial. These data are critical for integrating participants' views into future precision medicine delivery.

Long-term care for people treated for cancer during childhood and adolescence.

Worldwide advances in treatment and supportive care for children and adolescents with cancer have resulted in a increasing population of survivors growing into adulthood. Yet, this population is at very high risk of late occurring health problems, including significant morbidity and early mortality. Unique barriers to high-quality care for this group include knowledge gaps among both providers and survivors as well as fragmented health-care delivery during the transition from paediatric to adult care settings. Survivors of childhood and adolescent cancer are at risk for a range of late-occuring side-effects from treatment, including cardiac, endocrine, pulmonary, fertility, renal, psychological, cognitive, and socio-developmental impairments. Care coordination and transition to adult care are substantial challenges, but can be empowering for survivors and improve outcomes, and could be facilitated by clear, effective communication and support for self-management. Resources for adult clinical care teams and primary care providers include late-effects surveillance guidelines and web-based support services.

Childhood Cancer Survivors' Reported Late Effects, Motivations for Seeking Survivorship Care, and Patterns of Attendance.

BACKGROUND: Childhood cancer survivors are vulnerable to long-term treatment-related health conditions, which can lead to poor quality of life. Little data exist on the overall health of long-term Australian and New Zealand childhood cancer survivors or on survivors' motivations for attending survivorship clinics. METHODS: This study administers a cross-sectional questionnaire to long-term survivors ≥5 years from their primary diagnosis. We compared participant-reported number of late effects by a cancer diagnosis, and identified clinical (eg, treatment) and demographic (eg, age) factors that were associated with late effects burden and engagement in cancer survivorship care. RESULTS: A total of 634 participants completed questionnaires (48% male, mean age = 21.7 years). Most participants (79%) reported at least one cancer-related late effect, most commonly fatigue (40%) and memory/learning difficulties (34%). Brain tumor survivors reported a higher total number of late effects than survivors with other diagnoses (mean = 5.7 vs. 3.2, P < .001). Participants' most commonly reported motivators for engaging in care were to understand problems that may occur later in life because of their cancer and/or treatment (98.5%) and to get reassurance about one's health (97.4%). The proportion of survivors endorsing each motivating factor was similar across cancer diagnoses, with the exception of learning more about insurance and pensions (highest in brain tumor survivors = 80%). In multivariable analyses, survivors were more likely to report being engaged in survivorship care if they were younger (P < .001), less time had elapsed since their diagnosis (P < .001), or they reported a higher number of motivating factors (P = .016). CONCLUSION: Survivors report a range of health problems decades after treatment completion. Understanding the burden of late effects, and motivators for seeking survivorship care to manage these health problems, is important for ensuring that tailored interventions or services are available to meet the needs of this growing population and to design effective models of survivorship care.

Child-centered communication interventions in pediatric oncology: A scoping review and proposed new communication model.

Child-centered communication in pediatric oncology can be challenging. We aimed to review communication interventions with children about cancer treatment and prognosis to identify potentially effective child-centered communication models and approaches. We updated a previous review on communication interventions in oncology and searched MEDLINE, Scopus, and PsychINFO for studies indexed between October 2019 up to October 2022. We further searched for ongoing studies on ClinicalTrials.gov. Communication interventions targeting pediatric oncology patients (below 18 years), with outcomes of communication, psychological symptoms or satisfaction in the target population were eligible. We identified 685 titles/abstracts, screened the full text of 34 studies and included only one published study and two ongoing studies. The published study tested a communication tool to help clinicians inform adolescents about treatment options and facilitate shared decision-making. No communication models were identified. We drew on knowledge from existing studies and guidelines to develop a new child-centered communication model.

Impact of Hospitalizations due to Chronic Health Conditions on Early Child Development.

OBJECTIVE: To assess the impact of hospitalization for chronic health conditions on early child development and wellbeing at school start. METHODS: We conducted a longitudinal cohort study of children starting school using population-based record linkage of routinely collected admitted hospital data and standardized assessment of early childhood development (Australian Early Developmental Census: AEDC). Developmental vulnerability (DV) was defined as children scoring <10th centile in any one of five developmental domains. Children scoring <10th centile on two or more domains were considered developmentally high-risk (DHR). Children hospitalized with chronic health conditions were compared to children without hospitalizations prior to school start. RESULTS: Among 152,851 children with an AEDC record, 22,271 (14·6%) were hospitalized with a chronic condition. Children hospitalized with chronic health conditions were more likely to be DHR (adjusted odds ratio 1.25, 95% CI: 1.18-1.31) compared to children without hospitalizations. Children hospitalized more frequently (>7 times) or with longer duration (>2 weeks) had a 40% increased risk of being DHR (1.40, 95% CI: 1.05-1.88 and 1.40, 95% CI: 1.13-1.74, respectively). Children hospitalized with mental health/behavioral/developmental conditions had the highest risk of DHR (2.23, 95% CI: 1.72-2.90). Developmental vulnerability was increased for physical health (1.37, 95% CI: 1.30-1.45), language (1.28, 95% CI: 1.19-1.38), social competence (1.22, 95% CI: 1.16-1.29), communication (1.17, 95% CI: 1.10-1.23), and emotional maturity (1.16, 95% CI: 1.09-1.23). CONCLUSIONS: Frequent and longer duration hospitalizations for chronic health conditions can impact early childhood development. Research and interventions are required to support future development and well-being of children with chronic health conditions who are hospitalized.

Acute Biological Changes in Gynecologic Surgeons during Surgery: A Prospective Study.

STUDY OBJECTIVE: To assess changes in biological measures of acute stress in surgeons during surgery in real-world settings DESIGN: A prospective cohort study. SETTING: A tertiary teaching hospital. PATIENTS: 8 consultant and 9 training gynecologists. INTERVENTION: A total of, 161 elective gynecologic surgeries of 3 procedures: laparoscopic hysterectomy, laparoscopic excision of endometriosis, or hysteroscopic myomectomy. MEASUREMENTS AND MAIN RESULTS: Changes in surgeons' biological measures of acute stress while undertaking elective surgery. Salivary cortisol, mean and maximum heart rate (HR), and indices of the HR variability were recorded before and during surgery. From baseline to during surgery over the cohort, salivary cortisol decreased from 4.1 nmol/L to 3.6 nmol/L (p = .03), maximum HR increased from 101.8 beats per min (bpm) to 106.5 bpm (p <.01), root mean square of standard deviation decreased from 51.1 ms to 39.0 ms (p <.01), and standard deviation of beat-to-beat variability decreased from 73.7 to 59.8 ms (p <.01). Analysis of individual changes in stress by participant-surgery event by paired data graphs reveal inconsistent direction of change in all measures of biological stress despite stratification by surgical experience, role in surgery, level of training, or type of surgery performed. CONCLUSION: This study measured biometric stress changes at both a group and individual level in real-world, live surgical settings. Individual changes have not previously been reported and the variable direction of stress change by participant-surgery episode identified in this study demonstrates a problematic interpretation of mean cohort findings previously reported. Results from this study suggest that either live surgery with tight environment control or surgical simulation studies may identify what, if any, biological measures of stress can predict acute stress reactions during surgery.

Recommendations for the surveillance of mental health problems in childhood, adolescent, and young adult cancer survivors: a report from the International Late Effects of Childhood Cancer Guideline Harmonization Group.

Survivors of childhood, adolescent, and young adult (diagnosed when <25 years of age) cancer are at risk of mental health problems. The aim of this clinical practice guideline is to harmonise international recommendations for mental health surveillance in survivors of childhood, adolescent, and young adult cancer. This guideline was developed by a multidisciplinary panel of experts under the sponsorship of the International Guideline Harmonization Group. We evaluated concordance among existing survivorship clinical practice guidelines and conducted a systematic review following evidence-based methods. Of 7249 studies identified, 76 articles from 12 countries met the inclusion criteria. Recommendations were formulated on the basis of identified evidence in combination with clinical considerations. This international clinical practice guideline strongly recommends mental health surveillance for all survivors of childhood, adolescent, and young adult cancers at every follow-up visit and prompt referral to mental health specialists when problems are identified. Overall, the recommendations reflect the necessity of mental health surveillance as part of comprehensive survivor-focused health care.

Prospective longitudinal evaluation of treatment-related toxicity and health-related quality of life during the first year of treatment for pediatric acute lymphoblastic leukemia.

BACKGROUND: Pediatric acute lymphoblastic leukemia (ALL) therapy is accompanied by treatment-related toxicities (TRTs) and impaired quality of life. In Australia and New Zealand, children with ALL are treated with either Children's Oncology Group (COG) or international Berlin-Frankfurt-Munster (iBFM) Study Group-based therapy. We conducted a prospective registry study to document symptomatic TRTs (venous thrombosis, neurotoxicity, pancreatitis and bone toxicity), compare TRT outcomes to retrospective TRT data, and measure the impact of TRTs on children's general and cancer-related health-related quality of life (HRQoL) and parents' emotional well-being. METHODS: Parents of children with newly diagnosed ALL were invited to participate in the ASSET (Acute Lymphoblastic Leukaemia Subtypes and Side Effects from Treatment) study and a prospective, longitudinal HRQoL study. TRTs were reported prospectively and families completed questionnaires for general (Healthy Utility Index Mark 3) and cancer specific (Pediatric Quality of Life Inventory (PedsQL)-Cancer Module) health related quality of life as well the Emotion Thermometer to assess emotional well-being. RESULTS: Beginning in 2016, 260 pediatric patients with ALL were enrolled on the TRT registry with a median age at diagnosis of 59 months (range 1-213 months), 144 males (55.4%), majority with Pre-B cell immunophenotype, n = 226 (86.9%), 173 patients (66.5%) treated according to COG platform with relatively equal distribution across risk classification sub-groups. From 2018, 79 families participated in the HRQoL study through the first year of treatment. There were 74 TRT recorded, reflecting a 28.5% risk of developing a TRT. Individual TRT incidence was consistent with previous studies, being 7.7% for symptomatic VTE, 11.9% neurotoxicity, 5.4% bone toxicity and 5.0% pancreatitis. Children's HRQoL was significantly lower than population norms throughout the first year of treatment. An improvement in general HRQoL, measured by the HUI3, contrasted with the lack of improvement in cancer-related HRQoL measured by the PedsQL Cancer Module over the first 12 months. There were no persisting differences in the HRQoL impact of COG compared to iBFM therapy. CONCLUSIONS: It is feasible to prospectively monitor TRT incidence and longitudinal HRQoL impacts during ALL therapy. Early phases of ALL therapy, regardless of treatment platform, result in prolonged reductions in cancer-related HRQoL.

'Like ships in the night': A qualitative investigation of the impact of childhood cancer on parents' emotional and sexual intimacy.

BACKGROUND: Childhood cancer is highly distressing for families and can place strain on parents' relationships. Parental functioning and cohesiveness are important predictors of family functioning and adaptation to stress. This qualitative study investigated the perceived impact of childhood cancer on parents' relationship with their partner, with a focus on emotional and sexual intimacy. METHODS: We conducted semi-structured interviews with 48 parents (42 mothers, six fathers) of children under the age of 18 who had completed curative cancer treatment. We analysed the interviews using thematic analysis. RESULTS: At interview, parents were on average 40.7 years old (SD = 5.5, range: 29-55 years), and had a child who had completed cancer treatment between 3 months and 10.8 years previously (M = 22.1 months). All participants were living with their partner in a married/de facto relationship. Most parents reported that their child's cancer treatment had a negative impact on emotional and sexual intimacy with their partner, with some impacts extending to the post-treatment period. Reasons for compromised intimacy included exhaustion and physical constraints, having a shifted focus, and discord arising from different coping styles. Some parents reported that their relationship strengthened. Parents also discussed the impact of additional stressors unrelated to the child's cancer experience. CONCLUSIONS: Parents reported that childhood cancer had a negative impact on aspects of emotional and sexual intimacy, although relationship strengthening was also evident. It is important to identify and offer support to couples who experience ongoing relationship stress, which may have adverse effects on family functioning and psychological wellbeing into survivorship.

Poor diet quality and adverse eating behaviors in young survivors of childhood cancer.

BACKGROUND: The long-term impact of childhood cancer treatment on dietary intake is likely to be complex, and the length of time dietary behaviors are affected after childhood cancer treatment is unknown. AIM: The aim of this study was to determine the diet quality in childhood cancer survivors recently off treatment and explore possible contributing factors that may affect diet quality in this population. METHODS: Participants were 65 parents and/or carers of childhood cancer survivors (CCS) (aged 2-18 years), recently off treatment, and 81 age-matched controls. Participants completed two self-administered dietary intake and eating behavior questionnaires. Study data were explored to determine between group differences, bivariate analysis using Spearman correlations was used to determine the relationship between diet quality and identified variables, and hierarchical cluster analysis was completed to characterize specific variables into clusters. RESULTS: One hundred and forty-six parents of children aged 2-18 years completed the study (65 parents of CCS and 81 control). CCS had a significantly poorer diet quality score than the age-matched controls (32.25 vs 34.83, P = 0.028). CCS had significantly higher parent-reported rates of "picky eating" behavior than the control group (2.31 vs 1.91; P = 0.044). Factors such as picky eating, emotional overeating, and body mass index z-score might drive diet quality in survivors. CONCLUSIONS: CCS were found to have poorer diet quality and more likely to have parents report picky eating behaviors. The outcomes highlighted the need for a tailored intervention aimed at improving healthy eating behaviors in CCS after treatment for cancer.

Inequality in early childhood chronic health conditions requiring hospitalisation: A data linkage study of health service utilisation and costs.

BACKGROUND: The cost of socioeconomic inequality in health service use among Australian children with chronic health conditions is poorly understood. OBJECTIVES: To quantify the cost of socioeconomic inequality in health service use among Australian children with chronic health conditions. METHODS: Cohort study using a whole-of-population linked administrative data for all births in Queensland, Australia, between July 2015 and July 2018. Socioeconomic status was defined by an areas-based measure, grouping children into quintiles from most disadvantaged (Q1) to least disadvantaged (Q5) based on their postcode at birth. Study outcomes included health service utilisation (inpatient, emergency department, outpatient, general practitioner, specialist, pathology and diagnostic imaging services) and healthcare costs. RESULTS: Of the 238,600 children included in the analysis, 10.4% had at least one chronic health condition. Children with chronic health conditions in Q1 had higher rates of inpatient (6.6, 95% confidence interval [CI] 6.4, 6.7), emergency department (7.2, 95% CI 7.0, 7.5) and outpatient (20.3, 95% CI 19.4, 21.3) service use compared to children with chronic health conditions in Q5. They also had lower rates of general practitioner (37.5, 95% CI 36.7, 38.4), specialist (8.9, 95% CI 8.5, 9.3), pathology (10.7, 95% CI 10.2, 11.3), and diagnostic imaging (4.3, 95% CI 4.2,4.5) service use. Children with any chronic health condition in Q1 incurred lower median out-of-pocket fees than children in Q5 ($0 vs $741, respectively), lower median Medicare funding ($2710, vs $3408, respectively), and higher median public hospital funding ($31, 052 vs $23, 017, respectively). CONCLUSIONS: Children of most disadvantage are more likely to access public hospital provided services, which are accessible free of charge to patients. These children are less likely to access general practitioner, specialist, pathology and diagnostic imaging services; all of which are critical to the ongoing management of chronic health conditions, but often attract an out-of-pocket fee.