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Background Early efforts at risk-adapted therapy for neuroblastoma are predicted to result in differential late effects; the magnitude of these differences has not been well described. Methods Late mortality, subsequent malignant neoplasms (SMNs), and severe/life-threatening chronic health conditions (CHCs), graded according to CTCAE v4.03, were assessed among 5-year Childhood Cancer Survivor Study (CCSS) survivors of neuroblastoma diagnosed 1987-1999. Using age, stage at diagnosis, and treatment, survivors were classified into risk groups (low [n = 425]; intermediate [n = 252]; high [n = 245]). Standardized mortality ratios (SMRs) and standardized incidence ratios (SIRs) of SMNs were compared with matched population controls. Cox regression models estimated hazard ratios (HRs) and 95% confidence intervals for CHC compared with 1029 CCSS siblings. Results Among survivors (49.8% male; median age = 21 years, range = 7-42; median follow-up = 19.3 years, range = 5-29.9), 80% with low-risk disease were treated with surgery alone, whereas 79.1% with high-risk disease received surgery, radiation, chemotherapy ± autologous stem cell transplant (ASCT). All-cause mortality was elevated across risk groups (SMRhigh = 27.7 [21.4-35.8]; SMRintermediate = 3.3 [1.7-6.5]; SMRlow = 2.8 [1.7-4.8]). SMN risk was increased among high- and intermediate-risk survivors (SIRhigh = 28.0 [18.5-42.3]; SIRintermediate = 3.7 [1.2-11.3]) but did not differ from the US population for survivors of low-risk disease. Compared with siblings, survivors had an increased risk of grade 3-5 CHCs, particularly among those with high-risk disease (HRhigh = 16.1 [11.2-23.2]; HRintermediate = 6.3 [3.8-10.5]; HRlow = 1.8 [1.1-3.1]). Conclusion Survivors of high-risk disease treated in the early days of risk stratification carry a markedly elevated burden of late recurrence, SMN, and organ-related multimorbidity, whereas survivors of low/intermediate-risk disease have a modest risk of late adverse outcomes. [ABSTRACT FROM AUTHOR]

Background: Adolescents with cancer can receive care in pediatric or adult institutions. Survival often differs by locus, but little is known about relative health care utilization and costs. We estimated these in a population-based cohort of adolescents.Methods: All Ontario adolescents (15.0-17.9 years) diagnosed with cancer between 1995 and 2010 were identified from provincial cancer registries. We compared health care resource utilization (hospitalizations, emergency department visits, same-day surgeries, outpatient chemotherapy, radiation, diagnostic/laboratory tests, physician services, home care) and costs (2012 Canadian dollars) during four discrete care phases-prediagnosis (60 days), initial (360 days), continuing (variable), and terminal (360 days)-between adolescents treated in pediatric vs adult institutions, for the whole cohort and within seven diagnostic categories. All statistical tests were two-sided.Results: Of 1356 eligible adolescents, 691 and 665 were treated in adult and pediatric institutions, respectively. Hospitalization rates were higher in pediatric institutions during prediagnosis (14.9% vs 6.9%, P < .001), initial (95.1% vs 73.3%, P < .001), and continuing phases (43.2% vs 34.4%, P = .002), but similar (96.1% vs 96.3%, P = .93) during the terminal phase. Average length of stay was higher at pediatric institutions within most diagnoses and phases. For all diagnoses, median initial phase costs were higher in pediatric than adult institutions (eg, leukemia: $153 926 vs $102 418 per 360 days, P < .001; lymphoma: $65 025 vs $19 846, P < .001, respectively).Conclusions: The costs of caring for adolescents with the same malignancy are considerably higher in pediatric than adult institutions during most phases. Resource utilization, particularly hospitalization, drives much of the cost difference, making these data applicable to other jurisdictions. [ABSTRACT FROM AUTHOR]

Background: The 5-year survival rate for individuals with neuroblastoma is approaching 70%. Few data exist, however, on the long-term outcomes of these patients, who are often treated at a very young age. Methods: Outcome data were obtained for 954 5-year neuroblastoma survivors who were diagnosed in 1970-1986 and enrolled in the Childhood Cancer Survivor Study (CCSS). Late mortality, second malignant neo-plasms, and chronic health conditions were analyzed in relation to treatment factors using Poisson regression models and their modification with generalized estimating equations. Neuroblastoma survivors were compared with a cohort of 3899 siblings of CCSS participants for risk of chronic health conditions and selected sociodemographic outcomes. All statistical tests were two-sided. Results: Six percent of patients died more than 5 years after their diagnosis (standardized mortality ratio = 5.6; 95% confidence interval [l] = 4.4 to 6.9). The most common causes of death were disease recurrence (n = 43) and second malignant neoplasms (n = 13). The cumulative incidence of second malignant neoplasms was 3.5% at 25 years and 7.0% at 30 years after diagnosis. Compared with the sibling cohort, survivors had an increased risk of selected chronic health conditions (risk ratio ERR) = 8.3; 95% Cl = 7.1 to 9.7) with a 20-year cumulative incidence of 41.1%. The most prevalent outcomes involved the neurological, sensory, endocrine, and musculoskeletal systems, with 20-year cumulative incidences of 29.8%, 8.6%, 8.3%, and 7.8%, respectively. Neuroblastoma survivors who were treated with multimodality therapy were more likely to develop a chronic health condition than survivors treated with surgery alone (RR = 2.2; 95% Cl = 1.6 to 3.0). Neuroblastoma survivors were less likely than siblings to have ever been employed (P= .04) or to be married (P< .001) and had a lower-personal income (P= .009). Conclusions: Neuroblastoma survivors have an increased rate of mortality and second malignant neoplasms, relative to the age- and sex-comparable US population, and of chronic health conditions, relative to their siblings, which underscores the need for long-term medical surveillance. [ABSTRACT FROM AUTHOR]