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- Title
Long-term Outcomes in Survivors of Neuroblastoma: A Report From the Childhood Cancer Survivor Study.
- Authors
Laverdiëre, Caroline; Liu, Qi; Yasui, Yutaka; Nathan, Paul C.; Gurney, James G.; Stovall, Marilyn; Diller, Lisa R.; Cheung, Nai-Kong; Wolden, Suzanne; Robison, Leslie L.; Sklar, Charles A.
- Abstract
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.
- Subjects
NEUROBLASTOMA; CHILDHOOD cancer; CHRONIC disease risk factors; POISSON processes; REGRESSION analysis; SOCIODEMOGRAPHIC factors; DISEASE incidence
- Publication
JNCI: Journal of the National Cancer Institute, 2009, Vol 101, Issue 16, p1131
- ISSN
0027-8874
- Publication type
Article
- DOI
10.1093/jnci/djp230