The Impact of Specialist Care on Teenage and Young Adult Patient-Reported Outcomes in England: A BRIGHTLIGHT Study.

Purpose: In England, health care policy promotes specialized age-appropriate cancer services for teenagers and young adults (TYA), for those aged 13-24 years at diagnosis. Specialist Principal Treatment Centers (PTCs) provide enhanced age-specific care for TYA, although many still receive all or some of their care in adult or children's cancer services. Our aim was to determine the patient-reported outcomes associated with TYA-PTC based care. Methods: We conducted a multicenter cohort study, recruiting 1114 TYA aged 13-24 years at diagnosis. Data collection involved a bespoke survey at 6,12,18, 24, and 36 months after diagnosis. Confounder adjusted analyses of perceived social support, illness perception, anxiety and depression, and health status, compared patients receiving NO-TYA-PTC care with those receiving ALL-TYA-PTC and SOME-TYA-PTC care. Results: Eight hundred and thirty completed the first survey. There was no difference in perceived social support, anxiety, or depression between the three categories of care. Significantly higher illness perception was observed in the ALL-TYA-PTC and SOME-TYA-PTC group compared to the NO-TYA-PTC group, (adjusted difference in mean (ADM) score on Brief Illness Perception scale 2.28 (95% confidence intervals [CI] 0.48-4.09) and 2.93 [1.27-4.59], respectively, p = 0.002). Similarly, health status was significantly better in the NO-TYA-PTC (ALL-TYA-PTC: ADM -0.011 [95%CI -0.046 to 0.024] and SOME-TYA-PTC: -0.054 [-0.086 to -0.023]; p = 0.006). Conclusion: The reason for the difference in perceived health status is unclear. TYA who accessed a TYA-PTC (all or some care) had higher perceived illness. This may reflect greater education and promotion of self-care by health care professionals in TYA units.

Delivered relative dose intensity in adolescent and young adult germ cell tumours in England: Assessment of data quality and consistency from clinical trials compared to national cancer registration data.

Adolescent and young adults (AYA) with germ cell tumours (GCT) have poorer survival rates than children and many older adults with the same cancers. There are several likely contributing factors to this, including the treatment received. The prognostic benefit of intended dose intensity is well documented in GCT from trials comparing regimens. However, evidence specific to AYA is limited by poor recruitment of AYA to trials and dose delivery outside trials not being well examined. We examined the utility of cancer registration data and a clinical trials dataset to investigate the delivery of relative dose intensity (RDI) in routine National Health Service practice in England, compared to within international clinical trials. Linked data from the Cancer Outcomes and Services Dataset (COSD) and the Systemic Anti-Cancer Therapy (SACT) dataset, and data from four international clinical trials were analysed. Survival over time was described using Kaplan-Meier estimation; overall, by age category, International Germ-Cell Cancer Collaborative Group (IGCCCG) classification, stage, tumour subtype, primary site, ethnicity and deprivation. Cox regression models were used to determine the fully adjusted effect of RDI on mortality risk. The quality of both datasets was critically evaluated and clinically enhanced. RDI was found to be well maintained in all datasets with higher RDIs associated with improved survival outcomes. Real-world data demonstrated several strengths, including population coverage and inclusion of sociodemographic variables and comorbidity. It is limited in GCT however, by the poor completion of data items enabling risk classification of patients and a higher proportion of missing data.

Patient and public involvement to inform priorities and practice for research using existing healthcare data for children's and young people's cancers.

BACKGROUND: In the United Kingdom, healthcare data is collected on all patients receiving National Health Service (NHS) care, including children and young people (CYP) with cancer. This data is used to inform service delivery, and with special permissions used for research. The use of routinely collected health data in research is an advancing field with huge potential benefit, particularly in CYP with cancer where case numbers are small and the impact across the life course can be significant. Patient and public involvement (PPI) exercise aims: Identify current barriers to trust relating to the use of healthcare data for research. Determine ways to increase public and patient confidence in the use of healthcare data in research. Define areas of research importance to CYP and their carers using healthcare data. METHODS: Young people currently aged between 16 and 25 years who had a cancer diagnosis before the age of 20 years and carers of a young person with cancer were invited to take part via social media and existing networks of service users. Data was collected during two interactive online workshops totalling 5 h and comprising of presentations from health data experts, case-studies and group discussions. With participant consent the workshops were recorded, transcribed verbatim and analysed using thematic analysis. RESULTS: Ten young people and six carers attended workshop one. Four young people and four carers returned for workshop two. Lack of awareness of how data is used, and negative media reporting were seen as the main causes of mistrust. Better communication and education on how data is used were felt to be important to improving public confidence. Participants want the ability to have control over their own data use. Late effects, social and education outcomes and research on rare tumours were described as key research priorities for data use. CONCLUSIONS: In order to improve public and patient trust in our use of data for research, we need to improve communication about how data is used and the benefits that arise.

Everyday data is collected on all patients treated within the National Health Service, including children and young people with cancer (CYP). This data is used routinely to improve how services are run and with special permissions, can also be used for research. Negative reporting in the media about this use of data can lead to mistrust and some people choosing not to share their data. This can reduce the quality and accuracy of research looking at rare diseases or populations with small numbers. In addition, many barriers exist to researchers when trying to access this data such as laws around data sharing, making it difficult and sometimes impossible to carry out such research. We invited CYP and carers to two workshops to: Learn about how healthcar e data is used for research. Consider ways to increase public and patient confidence in this use of healthcare data. Describe areas of research importance to CYP and their carers using healthcare data. Ten young people and six carers attended the first workshop. Four young people and four carers returned for workshop two. Workshops consisted of interactive presentations, case studies and group discussions. Overall participants felt that lack of awareness and negative media reporting led to mistrust in data use for research. It was believed that greater education about how the data is used, including positive examples of the benefits of the research, was needed to improve public confidence. Key research priorities for data use included late-effects, social and educational outcomes and rare tumours.

Regional perspectives on the coordination and delivery of paediatric end-of-life care in the UK: a qualitative study.

BACKGROUND: Provision of and access to paediatric end-of-life care is inequitable, but previous research on this area has focused on perspectives of health professionals in specific settings or children with specific conditions. This qualitative study aimed to explore regional perspectives of the successes, and challenges to the equitable coordination and delivery of end-of-life care for children in the UK. The study provides an overarching perspective on the challenges of delivering and coordinating end-of-life care for children in the UK, and the impact of these on health professionals and organisations. Previous research has not highlighted the successes in the sector, such as the formal and informal coordination of care between different services and sectors. METHODS: Semi-structured interviews with Chairs of the regional Palliative Care Networks across the UK. Chairs or co-Chairs (n = 19) of 15/16 Networks were interviewed between October-December 2021. Data were analysed using thematic analysis. RESULTS: Three main themes were identified: one standalone theme ("Communication during end-of-life care"); and two overarching themes ("Getting end-of-life services and staff in the right place", with two themes: "Access to, and staffing of end-of-life care" and "Inconsistent and insufficient funding for end-of-life care services"; and "Linking up healthcare provision", with three sub-themes: "Coordination successes", "Role of the networks", and "Coordination challenges"). Good end-of-life care was facilitated through collaborative and network approaches to service provision, and effective communication with families. The implementation of 24/7 advice lines and the formalisation of joint-working arrangements were highlighted as a way to address the current challenges in the specialism. CONCLUSIONS: Findings demonstrate how informal and formal relationships between organisations and individuals, enabled early communication with families, and collaborative working with specialist services. Formalising these could increase knowledge and awareness of end of life care, improve staff confidence, and overall improve professionals' experiences of delivering care, and families' experiences of receiving it. There are considerable positives that come from collaborative working between different organisations and sectors, and care could be improved if these approaches are funded and formalised. There needs to be consistent funding for paediatric palliative care and there is a clear need for education and training to improve staff knowledge and confidence.

Identifying the important social outcomes for childhood cancer survivors: an e-Delphi study protocol.

INTRODUCTION: Optimising the health of childhood cancer survivors is important given the high long-term survival rate coupled with a significant late effects burden. Included within the WHO's definition of 'Health' are social outcomes. These are of interest given their impact on adult functioning within society, complex interactions with physical and mental health outcomes and potential for cross generational effects. Categories included within the definition of social outcomes are ill defined leading to potential gaps in research and service provision which could affect the ability of survivors to achieve their maximal potential. An e-Delphi study will be used to achieve expert consensus on the most important social outcomes for childhood cancer survivors to inform future research and ultimately, service provision. METHODS AND ANALYSIS: A heterogeneous sample of at least 48 panel members will be recruited across four groups chosen to provide different perspectives on the childhood cancer journey: childhood cancer survivors, health professionals, social workers and teachers. Purposive sampling from a UK, regional long-term follow-up clinic will be used to recruit a representative sample of survivors. Other panel members will be recruited through local channels and national professional working groups. Opinions regarding breakdown and relevance of categories of social outcome will be collected through 3-5 rounds of questionnaires using an e-Delphi technique. Open ended, 7-point Likert scale and ranking questions will be used. Each round will be analysed collectively and per group to assess inter-rater agreement. Agreement and strength of agreement will be indicated by a median score of 6 or 7 and mean absolute deviation from the median, respectively. ETHICS AND DISSEMINATION: Ethical approval for this study has been granted by Regional Ethics Committee 4, West of Scotland (ID 297344). Study findings will be disseminated to involved stakeholders, published in a peer-reviewed journal and presented at conferences.

Linkage of National Congenital Heart Disease Audit data to hospital, critical care and mortality national data sets to enable research focused on quality improvement.

OBJECTIVES: To link five national data sets (three registries, two administrative) and create longitudinal healthcare trajectories for patients with congenital heart disease (CHD), describing the quality and the summary statistics of the linked data set. DESIGN: Bespoke linkage of record-level patient identifiers across five national data sets. Generation of spells of care defined as periods of time-overlapping events across the data sets. SETTING: National Congenital Heart Disease Audit (NCHDA) procedures in public (National Health Service; NHS) hospitals in England and Wales, paediatric and adult intensive care data sets (Paediatric Intensive Care Audit Network; PICANet and the Case Mix Programme from the Intensive Care National Audit & Research Centre; ICNARC-CMP), administrative hospital episodes (hospital episode statistics; HES inpatient, outpatient, accident and emergency; A&E) and mortality registry data. PARTICIPANTS: Patients with any CHD procedure recorded in NCHDA between April 2000 and March 2017 from public hospitals. PRIMARY AND SECONDARY OUTCOME MEASURES: Primary: number of linked records, number of unique patients and number of generated spells of care. Secondary: quality and completeness of linkage. RESULTS: There were 143 862 records in NCHDA relating to 96 041 unique patients. We identified 65 797 linked PICANet patient admissions, 4664 linked ICNARC-CMP admissions and over 6 million linked HES episodes of care (1.1M inpatient, 4.7M outpatient). The linked data set had 4 908 153 spells of care after quality checks, with a median (IQR) of 3.4 (1.8-6.3) spells per patient-year. Where linkage was feasible (in terms of year and centre), 95.6% surgical procedure records were linked to a corresponding HES record, 93.9% paediatric (cardiac) surgery procedure records to a corresponding PICANet admission and 76.8% adult surgery procedure records to a corresponding ICNARC-CMP record. CONCLUSIONS: We successfully linked four national data sets to the core data set of all CHD procedures performed between 2000 and 2017. This will enable a much richer analysis of longitudinal patient journeys and outcomes. We hope that our detailed description of the linkage process will be useful to others looking to link national data sets to address important research priorities.

The incidence and spectrum of congenital hand differences in patients with Fanconi anaemia: analysis of 48 patients.

We analysed the spectrum of congenital hand differences in a cohort of patients with Fanconi anaemia (FA). Data of 48 FA patients at the National Cancer Institute were reviewed focusing on age at diagnosis, type and severity of limb difference and any potential association with other known clinical anomalies that are part of the FA phenotype, specifically VACTERL-H and PHENOS. Twenty-eight patients had an upper limb difference, which always included thumb hypoplasia. Twenty-three patients had bilateral upper limb differences, including varying combinations and severities of thumb hypoplasia, radial dysplasia and thumb duplication. Patients with a limb difference were diagnosed at a younger age (<2 years: 15/28 with limb anomaly versus 4/20 without a limb anomaly). However, 7/28 with limb anomalies, usually thumb hypoplasia, were not diagnosed until after 6 years of age. This study demonstrates the broad spectrum of radial ray anomalies within the FA phenotype along with the possibility of either unilateral or bilateral upper limb differences and adds further merit to consideration of screening for FA in all cases of radial ray anomaly.Level of evidence: II.

International variation in childhood cancer mortality rates from 2001 to 2015: Comparison of trends in the International Cancer Benchmarking Partnership countries.

Despite improved survival rates, cancer remains one of the most common causes of childhood death. The International Cancer Benchmarking Partnership (ICBP) showed variation in cancer survival for adults. We aimed to assess and compare trends over time in cancer mortality between children, adolescents and young adults (AYAs) and adults in the six countries involved in the ICBP: United Kingdom, Denmark, Australia, Canada, Norway and Sweden. Trends in mortality between 2001 and 2015 in the six original ICBP countries were examined. Age standardised mortality rates (ASR per million) were calculated for all cancers, leukaemia, malignant and benign central nervous system (CNS) tumours, and non-CNS solid tumours. ASRs were reported for children (age 0-14 years), AYAs aged 15 to 39 years and adults aged 40 years and above. Average annual percentage change (AAPC) in mortality rates per country were estimated using Joinpoint regression. For all cancers combined, significant temporal reductions were observed in all countries and all age groups. However, the overall AAPC was greater for children (-2.9; 95% confidence interval = -4.0 to -1.7) compared to AYAs (-1.8; -2.1 to -1.5) and adults aged >40 years (-1.5; -1.6 to -1.4). This pattern was mirrored for leukaemia, CNS tumours and non-CNS solid tumours, with the difference being most pronounced for leukaemia: AAPC for children -4.6 (-6.1 to -3.1) vs AYAs -3.2 (-4.2 to -2.1) and over 40s -1.1 (-1.3 to -0.8). AAPCs varied between countries in children for all cancers except leukaemia, and in adults over 40 for all cancers combined, but not in subgroups. Improvements in cancer mortality rates in ICBP countries have been most marked among children aged 0 to 14 in comparison to 15 to 39 and over 40 year olds. This may reflect better care, including centralised service provision, treatment protocols and higher trial recruitment rates in children compared to older patients.

Oral etoposide as a single agent in childhood and young adult cancer in England: Still a poorly evaluated palliative treatment.

BACKGROUND: Oral etoposide is commonly used in palliative treatment of childhood and young adult cancer without robust evidence. We describe a national, unselected cohort of young people in England treated with oral etoposide using routinely collected, population-level data. METHODS: Patients aged under 25 years at cancer diagnosis (1995-2017) with a treatment record of single-agent oral etoposide in the Systemic AntiCancer Dataset (SACT, 2012-2018) were identified, linked to national cancer registry data using NHS number and followed to 5 January 2019. Overall survival (OS) was estimated for all tumours combined and by tumour group. A Cox model was applied accounting for age, sex, tumour type, prior and subsequent chemotherapy. RESULTS: Total 115 patients were identified during the study period. Mean age was 11.8 years at cancer diagnosis and 15.5 years at treatment with oral etoposide. Median OS was 5.5 months from the start of etoposide; 13 patients survived beyond 2 years. Survival was shortest in patients with osteosarcoma (median survival 3.6 months) and longest in CNS embryonal tumours (15.5 months). Across the cohort, a median of one cycle (range one to nine) of etoposide was delivered. OS correlated significantly with tumour type and prior chemotherapy, but not with other variables. CONCLUSIONS: This report is the largest series to date of oral etoposide use in childhood and young adult cancer. Most patients treated in this real world setting died quickly. Despite decades of use, there are still no robust data demonstrating a clear benefit of oral etoposide for survival.

Risk stratification of young adult survivors of cancer to estimate hospital morbidity burden: applicability of a pediatric therapy-based approach.

PURPOSE: Children and young adults (CYA) are at risk of late morbidity following cancer treatment, with risk varying by disease type and treatment received. Risk-stratified levels of aftercare which stratify morbidity burden to inform the intensity of long-term follow-up care, are well established for survivors of cancer under the age of 18 years, utilizing the National Cancer Survivor Initiative (NCSI) approach. We investigated the applicability of risk-stratified levels of aftercare in predicting long-term morbidity in young adults (YA), aged 18-29 years. METHODS: Long-term CYA survivors followed-up at a regional center in the North of England were risk-stratified by disease and treatments received into one of three levels. These data were linked with local cancer registry and administrative health data (Hospital Episode Statistics), where hospital activity was used as a marker of late morbidity burden. RESULTS: Poisson modelling with incident rate ratios (IRR) demonstrated similar trends in hospital activity for childhood (CH) and YA cancer survivors across NCSI risk levels. NCSI levels independently predicted long-term hospitalization risk in both CH and YA survivors. Risk of hospitalization was significantly reduced for levels 1 (CH IRR 0.32 (95% CI 0.26-0.41), YA IRR 0.06 (95% CI 0.01-0.43)) and 2; CH IRR 0.46 (95% CI 0.42-0.50), YA IRR 0.49 (95% CI 0.37-0.50)), compared with level 3. CONCLUSIONS: The NCSI pediatric late-effects risk stratification system can be effectively and safely applied to cancer patients aged 18-29, independent of ethnicity or socioeconomic position. IMPLICATIONS FOR CANCER SURVIVORS: To enhance quality of care and resource utilization, long-term aftercare of survivors of YA cancer can and should be risk stratified through adoption of approaches such as the NCSI risk-stratification model.

Severity of COVID-19 in children with cancer: Report from the United Kingdom Paediatric Coronavirus Cancer Monitoring Project.

BACKGROUND: Children with cancer are frequently immunocompromised. While children are generally thought to be at less risk of severe SARS-CoV-2 infection than adults, comprehensive population-based evidence for the risk in children with cancer is unavailable. We aimed to produce evidence of the incidence and outcomes from SARS-CoV-2 in children with cancer attending all hospitals treating this population across the UK. METHODS: Retrospective and prospective observational study of all children in the UK under 16 diagnosed with cancer through data collection from all hospitals providing cancer care to this population. Eligible patients tested positive for SARS-CoV-2 on reverse transcription polymerase chain reaction (RT-PCR). The primary end-point was death, discharge or end of active care for COVID-19 for those remaining in hospital. RESULTS: Between 12 March 2020 and 31 July 2020, 54 cases were identified: 15 (28%) were asymptomatic, 34 (63%) had mild infections and 5 (10%) moderate, severe or critical infections. No patients died and only three patients required intensive care support due to COVID-19. Estimated incidence of hospital identified SARS-CoV-2 infection in children with cancer under 16 was 3%. CONCLUSIONS: Children with cancer with SARS-CoV-2 infection do not appear at increased risk of severe infection compared to the general paediatric population. This is reassuring and supports the continued delivery of standard treatment.

Risk of cerebrovascular disease among 13 457 five-year survivors of childhood cancer: A population-based cohort study.

Survivors of childhood cancer treated with cranial irradiation are at risk of cerebrovascular disease (CVD), but the risks beyond age 50 are unknown. In all, 13457 survivors of childhood cancer included in the population-based British Childhood Cancer Survivor Study cohort were linked to Hospital Episode Statistics data for England. Risk of CVD related hospitalisation was quantified by standardised hospitalisation ratios (SHRs), absolute excess risks and cumulative incidence. Overall, 315 (2.3%) survivors had been hospitalised at least once for CVD with a 4-fold risk compared to that expected (95% confidence interval [CI]: 3.7-4.3). Survivors of a central nervous system (CNS) tumour and leukaemia treated with cranial irradiation were at greatest risk of CVD (SHR = 15.6, 95% CI: 14.0-17.4; SHR = 5.4; 95% CI: 4.5-6.5, respectively). Beyond age 60, on average, 3.1% of CNS tumour survivors treated with cranial irradiation were hospitalised annually for CVD (0.4% general population). Cumulative incidence of CVD increased from 16.0% at age 50 to 26.0% at age 65 (general population: 1.4-4.2%). In conclusion, among CNS tumour survivors treated with cranial irradiation, the risk of CVD continues to increase substantially beyond age 50 up to at least age 65. Such survivors should be: counselled regarding this risk; regularly monitored for hypertension, dyslipidaemia and diabetes; advised on life-style risk behaviours. Future research should include the recall for counselling and brain MRI to identify subgroups that could benefit from pharmacological or surgical intervention and establishment of a case-control study to comprehensively determine risk-factors for CVD.

Cumulative burden of subsequent neoplasms, cardiovascular and respiratory morbidity in young people surviving cancer.

BACKGROUND: Long-term childhood and young adult cancer survivors are at increased risk of the late effects of multiple chronic conditions. In this study we estimate the cumulative burden of subsequent malignant neoplasms (SMN), cardiovascular and respiratory hospitalisations in long-term survivors of childhood and young adult cancers and associated treatment risks. METHODS: Five-year survivors of cancer diagnosed aged 0-29 years between 1992-2009 in Yorkshire, UK were included. The cumulative count of all hospital admissions (including readmissions) for cardiovascular and respiratory conditions and all SMNs diagnosed up to 2015 was calculated, with death as a competing risk. Associations between treatment exposures and cumulative burden were investigated using multiple-failure time survival models. RESULTS: A total of 3464 5-year survivors were included with a median follow-up of 8.2 years (IQR 4-13 years). Ten-years post diagnosis, the cumulative incidence for a respiratory admission was 6.0 % (95 %CI 5.2-6.9), a cardiovascular admission was 2.0 % (95 %CI 1.5-2.5), and SMN was 1.0 % (95 % CI 0.7-1.4) with an average of 13 events per 100 survivors observed (95 %CI 11-15). The risk of experiencing multiple events was higher for those treated with chemotherapy drugs with known lung toxicity (HR = 1.35, 95 %CI 1.09-1.68). DISCUSSION: Survivors of childhood and young adult cancer experience a high burden of morbidity due to respiratory, cardiovascular diseases and SMNs up to 20-years post-diagnosis. Statistical methods that capture multiple morbidities and recurrent events are important when quantifying the burden of late effects in young cancer survivors.

The Association Between Childhood Leukemia and Population Mixing: An Artifact of Focusing on Clusters?

BACKGROUND: Studies investigating the population-mixing hypothesis in childhood leukemia principally use two analytical approaches: (1) nonrandom selection of areas according to specific characteristics, followed by comparisons of their incidence of childhood leukemia with that expected based on the national average; and (2) regression analyses of region-wide data to identify characteristics associated with the incidence of childhood leukemia. These approaches have generated contradictory results. We compare these approaches using observed and simulated data. METHODS: We generated 10,000 simulated regions using the correlation structure and distributions from a United Kingdom dataset. We simulated cases using a Poisson distribution with the incidence rate set to the national average assuming the null hypothesis that only population size drives the number of cases. Selection of areas within each simulated region was based on characteristics considered responsible for elevated infection rates (population density and inward migration) and/or elevated leukemia rates. We calculated effect estimates for 10,000 simulations and compared results to corresponding observed data analyses. RESULTS: When the selection of areas for analysis is based on apparent clusters of childhood leukemia, biased assessments occur; the estimated 5-year incidence of childhood leukemia ranged between zero and eight per 10,000 children in contrast to the simulated two cases per 10,000 children, similar to the observed data. Performing analyses on region-wide data avoids these biases. CONCLUSIONS: Studies using nonrandom selection to investigate the association between childhood leukemia and population mixing are likely to have generated biased findings. Future studies can avoid such bias using a region-wide analytical strategy. See video abstract at, http://links.lww.com/EDE/B431.

Respiratory morbidity in young people surviving cancer: Population-based study of hospital admissions, treatment-related risk factors and subsequent mortality.

Respiratory diseases are a major cause of late morbidity and mortality amongst childhood cancer survivors. This population-based study investigates respiratory hospital admissions in long-term survivors of cancers diagnosed in young people to identify specific respiratory morbidities, treatment-related risks and their relationship to subsequent morbidity and mortality. Population-based cancer registrations in Yorkshire, England, diagnosed between 1990 and 2011 aged 0-29 years, were linked to inpatient Hospital Episode Statistics (HES) for admissions up to 2017. All 5-year survivors were included in analysis (n = 4235). Admission rates were compared to age- and sex- matched general population rates. Competing risk regression models were used to assess associations between treatment exposures and risk of admission. Risk of death after admission was calculated using Cox regression. By age 40, cumulative incidence for an admission for any type of respiratory condition was 49%. Respiratory admission rates were 1.86 times higher in cancer survivors than in the general population (95% Confidence Interval (CI) 1.73-2.01), and varied by respiratory condition and age at diagnosis. Treatment with chemotherapy with known lung toxicity increased the risk of admission for all respiratory conditions (subdistribution Hazard ratio (sHR) = 1.26, 95%CI 1.03-1.53) and pneumonia (sHR = 1.48, 95%CI 1.01-2.17). Subsequent mortality was highest in those admitted for pneumonia compared to other respiratory conditions (28% and 15% respectively). Survivors of childhood and young adult cancer remain at significantly increased risk of respiratory complications several decades after treatment, emphasising the importance for clinical initiatives for prevention, early detection and treatment.

Long-term survival after childhood acute lymphoblastic leukaemia: population-based trends in cure and relapse by clinical characteristics.

'Cure models' offer additional information to traditional epidemiological approaches to assess survival for cancer patients by simultaneously estimating the proportion cured and the survival of those 'uncured'. The proportion cured is a summary of long-term survival while the median survival time of the uncured provides important information on those who are not long-term survivors. Population-based trends in the cure proportion and survival of the uncured for childhood acute lymphoblastic leukaemia (ALL) by clinical prognostic risk factors were estimated using flexible parametric cure models, based on overall survival and event-free survival. Children aged 1-17 years diagnosed between 1990 and 2011 in Yorkshire, UK, were included (n = 492). The percentage cured increased from 77% (95% confidence interval 70-84%) in 1990-1997 to 89% (84-93%) in 2003-2011, while the median survival time of the uncured decreased from 3·2 years (2·2-4·1 years) to 0·7 years (0-1·5 years). Models based on event-free survival showed a similar trend. The 5-year cumulative incidence of relapse substantially decreased from 35% in 1990-97 to 9% in 2003-2011. These results show selective improvement in survival between 1990 and 2011 with a significant reduction in the risk of relapse alongside a reduced absolute duration of survival for those destined to be uncured.

Mental health of long-term survivors of childhood and young adult cancer: A systematic review.

Childhood cancer is increasing in prevalence whilst survival rates are improving. The prevalence of adult survivors of childhood cancer is consequently increasing. Many survivors suffer long-term consequences of their cancer treatment. Whilst many of these are well documented, relatively little is known about the mental health of survivors of childhood cancer. This article aimed to describe the prevalence and spectrum of mental health problems found in adult survivors of childhood cancer using a systematic review methodology. Our review included 67 articles, describing a number of problems, including depression, anxiety, behavioural problems and drug misuse. Factors increasing the likelihood of mental health problems included treatment with high-dose anthracyclines, cranial irradiation, diagnoses of sarcoma or central nervous system tumours and ongoing physical ill health. There were numerous limitations to the studies we found, including use of siblings of survivors as a control group, self-report methodology and lack of indications for prescriptions when prescribing data were used. This review has identified many mental health problems experienced by survivors of childhood cancer; however, the exact incidence, prevalence and risk-factors for their development remain unclear. Further work to identify childhood cancer patients who are at risk of developing late mental health morbidity is essential.