Dataset Information

Long-term inpatient disease burden in the Adult Life after Childhood Cancer in Scandinavia (ALiCCS) study: A cohort study of 21,297 childhood cancer survivors.

ABSTRACT:

Background

Survivors of childhood cancer are at increased risk for a wide range of late effects. However, no large population-based studies have included the whole range of somatic diagnoses including subgroup diagnoses and all main types of childhood cancers. Therefore, we aimed to provide the most detailed overview of the long-term risk of hospitalisation in survivors of childhood cancer.

Methods and findings

From the national cancer registers of Denmark, Finland, Iceland, and Sweden, we identified 21,297 5-year survivors of childhood cancer diagnosed with cancer before the age of 20 years in the periods 1943-2008 in Denmark, 1971-2008 in Finland, 1955-2008 in Iceland, and 1958-2008 in Sweden. We randomly selected 152,231 population comparison individuals matched by age, sex, year, and country (or municipality in Sweden) from the national population registers. Using a cohort design, study participants were followed in the national hospital registers in Denmark, 1977-2010; Finland, 1975-2012; Iceland, 1999-2008; and Sweden, 1968-2009. Disease-specific hospitalisation rates in survivors and comparison individuals were used to calculate survivors' standardised hospitalisation rate ratios (RRs), absolute excess risks (AERs), and standardised bed day ratios (SBDRs) based on length of stay in hospital. We adjusted for sex, age, and year by indirect standardisation. During 336,554 person-years of follow-up (mean: 16 years; range: 0-42 years), childhood cancer survivors experienced 21,325 first hospitalisations for diseases in one or more of 120 disease categories (cancer recurrence not included), when 10,999 were expected, yielding an overall RR of 1.94 (95% confidence interval [95% CI] 1.91-1.97). The AER was 3,068 (2,980-3,156) per 100,000 person-years, meaning that for each additional year of follow-up, an average of 3 of 100 survivors were hospitalised for a new excess disease beyond the background rates. Approximately 50% of the excess hospitalisations were for diseases of the nervous system (19.1% of all excess hospitalisations), endocrine system (11.1%), digestive organs (10.5%), and respiratory system (10.0%). Survivors of all types of childhood cancer were at increased, persistent risk for subsequent hospitalisation, the highest risks being those of survivors of neuroblastoma (RR: 2.6 [2.4-2.8]; n = 876), hepatic tumours (RR: 2.5 [2.0-3.1]; n = 92), central nervous system tumours (RR: 2.4 [2.3-2.5]; n = 6,175), and Hodgkin lymphoma (RR: 2.4 [2.3-2.5]; n = 2,027). Survivors spent on average five times as many days in hospital as comparison individuals (SBDR: 4.96 [4.94-4.98]; n = 422,218). The analyses of bed days in hospital included new primary cancers and recurrences. Of the total 422,218 days survivors spent in hospital, 47% (197,596 bed days) were for new primary cancers and recurrences. Our study is likely to underestimate the absolute overall disease burden experienced by survivors, as less severe late effects are missed if they are treated sufficiently in the outpatient setting or in the primary health care system.

Conclusions

Childhood cancer survivors were at increased long-term risk for diseases requiring inpatient treatment even decades after their initial cancer. Health care providers who do not work in the area of late effects, especially those in primary health care, should be aware of this highly challenged group of patients in order to avoid or postpone hospitalisations by prevention, early detection, and appropriate treatments.

SUBMITTER: de Fine Licht S

PROVIDER: S-EPMC5423554 | biostudies-literature | 2017 May

REPOSITORIES: biostudies-literature

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Long-term inpatient disease burden in the Adult Life after Childhood Cancer in Scandinavia (ALiCCS) study: A cohort study of 21,297 childhood cancer survivors.

de Fine Licht Sofie S Rugbjerg Kathrine K Gudmundsdottir Thorgerdur T Bonnesen Trine G TG Asdahl Peter Haubjerg PH Holmqvist Anna Sällfors AS Madanat-Harjuoja Laura L Tryggvadottir Laufey L Wesenberg Finn F Hasle Henrik H Winther Jeanette F JF Olsen Jørgen H JH

PLoS medicine 20170509 5

<h4>Background</h4>Survivors of childhood cancer are at increased risk for a wide range of late effects. However, no large population-based studies have included the whole range of somatic diagnoses including subgroup diagnoses and all main types of childhood cancers. Therefore, we aimed to provide the most detailed overview of the long-term risk of hospitalisation in survivors of childhood cancer.<h4>Methods and findings</h4>From the national cancer registers of Denmark, Finland, Iceland, and S ...[more]

PMID: 28486495

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Project description:BACKGROUND:Childhood cancer survivors are at risk for adverse psychological outcomes. Whether exercise can attenuate this risk is unknown. METHODS:In total, 6199 participants in the Childhood Cancer Survivor Study (median age, 34.3 years [range, 22.0-54.0 years]; median age at diagnosis, 10.0 years [range, 0-21.0 years]) completed a questionnaire assessing vigorous exercise and medical/psychological conditions. Outcomes were evaluated a median of 7.8 years (range, 0.1-10.0 years) later and were defined as: symptom level above the 90th percentile of population norms for depression, anxiety, or somatization on the Brief Symptom Inventory-18; cancer-related pain; cognitive impairment using a validated self-report neurocognitive questionnaire; or poor health-related quality of life. Log-binomial regression estimated associations between exercise (metabolic equivalent [MET]-hours per week-1 ) and outcomes adjusting for cancer diagnosis, treatment, demographics, and baseline conditions. RESULTS:The prevalence of depression at follow-up was 11.4% (95% CI, 10.6%-12.3%), anxiety 7.4% (95% CI, 6.7%-8.2%) and somatization 13.9% (95% CI, 13.0%-14.9%). Vigorous exercise was associated with lower prevalence of depression and somatization. The adjusted prevalence ratio for depression was 0.87 (95% CI, 0.72-1.05) for 3 to 6 MET hours per week-1 , 0.76 (95% CI, 0.62-0.94) for 9 to 12 MET-hours per week-1 , and 0.74 (95% CI, 0.58-0.95) for 15 to 21 MET-hours per week-1 . Compared with 0 MET hours per week-1 , 15 to 21 MET-hours per week-1 were associated with an adjusted prevalence ratio of 0.79 (95% CI, 0.62-1.00) for somatization. Vigorous exercise also was associated with less impairment in the physical functioning, general health and vitality (Ptrend < .001), emotional role limitations (Ptrend = .02), and mental health (Ptrend = .02) domains as well as higher cognitive function in the domains of task completion, organization, and working memory (P < .05 for all), but not in the domain of cancer pain. CONCLUSIONS:Vigorous exercise is associated with less psychological burden and cognitive impairment in childhood cancer survivors.

Project description:IntroductionThe growing number of survivors of childhood cancer, with many years of life ahead, demonstrates the increasing clinical and public health relevance of investigating the risks of social and socioeconomic impairment after a childhood cancer diagnosis and the life-saving treatment. To enrich understanding of the mental, social and socioeconomic difficulties that childhood cancer survivors may face during their life-course, identify particularly vulnerable survivors and overcome the limitations of previous research, we initiated the Socioeconomic Consequences in Adult Life after Childhood Cancer in Scandinavia (SALiCCS) research programme.MethodsThis Nordic cross-border research programme is a collaboration between the Danish Cancer Society, the Finnish Cancer Registry and Karolinska Institutet to investigate a broad range of mental, social and socioeconomic conditions in long-term childhood cancer survivors in Denmark, Finland and Sweden. SALiCCS is based on a registry-based matched cohort design, comprising five-year survivors of cancer diagnosed at ages 0-19 years (1971-2008 in Denmark, 1971-2009 in Finland, 1971-2011 in Sweden), age-, sex- and country-matched population comparisons and sibling comparisons who were followed over time. Outcomes of interest included mental disorders, educational achievements, employment and profession, family life and the need of social security benefits. Individual-level data linkage among various national registries provided the data for the research programme.ResultsThe SALiCCS core population comprises 21,292 five-year survivors, 103,303 population comparisons and 29,644 siblings as a second comparison group. The most common diagnoses in survivors were central nervous system tumours, leukaemias and lymphomas.DiscussionSALiCCS is the largest, most comprehensive population-based research initiative in this field, based on high-quality registry data with minimal risk of bias. The findings will be informative for evidence-based survivorship care targeting not only somatic late effects but also psychosocial impairments.

Project description:ImportanceAdvances in childhood and adolescent cancer treatment have been associated with increased rates of cure during the past 3 decades; however, improvement in adult life expectancy for these individuals has not yet been reported.ObjectivesTo project long-term survival and assess whether life expectancy will improve among adult survivors of childhood cancer who were treated in more recent decades.Design, setting, and participantsA microsimulation model of competing mortality risks was developed using data from the Childhood Cancer Survivor Study on 5-year survivors of childhood cancer diagnosed between 1970 and 1999. The model included (1) late recurrence, (2) treatment-related late effects (health-related [subsequent cancers, cardiac events, pulmonary conditions, and other] and external causes), and (3) US background mortality rates.ExposuresTreatment subgroups (no treatment or surgery only, chemotherapy alone, radiotherapy alone, and radiotherapy with chemotherapy) and individuals with acute lymphoblastic leukemia during childhood by era (1970-1979, 1980-1989, and 1990-1999).Main outcomes and measuresConditional life expectancy (defined as the number of years a 5-year survivor can expect to live), cumulative cause-specific mortality risk, and 10-year mortality risks conditional on attaining ages of 30, 40, 50, and 60 years.ResultsAmong the hypothetical cohort of 5-year survivors of childhood cancer representative of the Childhood Cancer Survivor Study participants (44% female and 56% male; mean [SD] age at diagnosis, 7.3 [5.6] years), conditional life expectancy was 48.5 years (95% uncertainty interval [UI], 47.6-49.6 years) for 5-year survivors diagnosed in 1970-1979, 53.7 years (95% UI, 52.6-54.7 years) for those diagnosed in 1980-1989, and 57.1 years (95% UI, 55.9-58.1 years) for those diagnosed in 1990-1999. Compared with individuals without a history of cancer, these results represented a gap in life expectancy of 25% (95% UI, 24%-27%) (16.5 years [95% UI, 15.5-17.5 years]) for those diagnosed in 1970-1979, 19% (95% UI, 17%-20%) (12.3 years [95% UI, 11.3-13.4 years]) for those diagnosed in 1980-1989, and 14% (95% UI, 13%-16%) (9.2 years [95% UI, 8.3-10.4 years]) for those diagnosed in 1990-1999. During the 3 decades, the proportion of survivors treated with chemotherapy alone increased (from 18% in 1970-1979 to 54% in 1990-1999), and the life expectancy gap in this chemotherapy-alone group decreased from 11.0 years (95% UI, 9.0-13.1 years) to 6.0 years (95% UI, 4.5-7.6 years). In contrast, during the same time frame, only modest improvements in the gap in life expectancy were projected for survivors treated with radiotherapy (21.0 years [95% UI, 18.5-23.2 years] to 17.6 years [95% UI, 14.2-21.2 years]) or with radiotherapy and chemotherapy (17.9 years [95% UI, 16.7-19.2 years] to 14.8 years [95% UI, 13.1-16.7 years]). For the largest group of survivors by diagnosis-those with acute lymphoblastic leukemia-the gap in life expectancy decreased from 14.7 years (95% UI, 12.8-16.5 years) in 1970-1979 to 8.0 years (95% UI, 6.2-9.7 years).Conclusions and relevanceEvolving treatment approaches are projected to be associated with improved life expectancy after treatment for pediatric cancer, in particular among those who received chemotherapy alone for their childhood cancer diagnosis. Despite improvements, survivors remain at risk for shorter lifespans, especially when radiotherapy was included as part of their childhood cancer treatment.

Project description:BackgroundAdult survivors of childhood central nervous system (CNS) malignancies are at high risk for long-term morbidity and late mortality. However, patterns of late mortality, the long-term risks of subsequent neoplasms and debilitating medical conditions, and sociodemographic outcomes have not been comprehensively characterized for individual diagnostic and treatment groups.MethodsWe collected information on treatment, mortality, chronic medical conditions, and neurocognitive functioning of adult 5-year survivors of CNS malignancies diagnosed between 1970 and 1986 within the Childhood Cancer Survivor Study. Using competing risk framework, we calculated cumulative mortality according to cause of death and cumulative incidence of subsequent neoplasms according to exposure and dose of cranial radiation therapy (RT). Neurocognitive impairment and socioeconomic outcomes were assessed with respect to dose of CNS radiotherapy to specific brain regions. Cumulative incidence of chronic medical conditions was compared between survivors and siblings using Cox regression models. All tests of statistical significance were two-sided.ResultsAmong all eligible 5-year survivors (n = 2821), cumulative late mortality at 30 years was 25.8% (95% confidence interval [CI] = 23.4% to 28.3%), due primarily to recurrence and/or progression of primary disease. Patients who received cranial RT of 50 Gy or more (n = 813) had a cumulative incidence of a subsequent neoplasm within the CNS of 7.1% (95% CI = 4.5% to 9.6%) at 25 years from diagnosis compared with 1.0% (95% CI = 0% to 2.3%) for patients who had no RT. Survivors had higher risk than siblings of developing new endocrine, neurological, or sensory complications 5 or more years after diagnosis. Neurocognitive impairment was high and proportional to radiation dose for specific tumor types. There was a dose-dependent association between RT to the frontal and/or temporal lobes and lower rates of employment, and marriage.ConclusionsSurvivors of childhood CNS malignancies are at high risk for late mortality and for developing subsequent neoplasms and chronic medical conditions. Care providers should be informed of these risks so they can provide risk-directed care and develop screening guidelines.

Dataset Information

Long-term inpatient disease burden in the Adult Life after Childhood Cancer in Scandinavia (ALiCCS) study: A cohort study of 21,297 childhood cancer survivors.

Background

Methods and findings

Conclusions

Publications

Long-term inpatient disease burden in the Adult Life after Childhood Cancer in Scandinavia (ALiCCS) study: A cohort study of 21,297 childhood cancer survivors.

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