Long-term morbidity and mortality among survivors of neuroblastoma diagnosed during infancy: A report from the Childhood Cancer Survivor Study Journal Article


Authors: Friedman, D. N.; Goodman, P. J.; Leisenring, W. M.; Diller, L. R.; Cohn, S. L.; Howell, R. M.; Smith, S. A.; Tonorezos, E. S.; Wolden, S. L.; Neglia, J. P.; Ness, K. K.; Gibson, T. M.; Nathan, P. C.; Weil, B. R.; Robison, L. L.; Oeffinger, K. C.; Armstrong, G. T.; Sklar, C. A.; Henderson, T. O.
Article Title: Long-term morbidity and mortality among survivors of neuroblastoma diagnosed during infancy: A report from the Childhood Cancer Survivor Study
Abstract: PURPOSETo describe the risk of late mortality, subsequent malignant neoplasms (SMNs), and chronic health conditions (CHCs) in survivors of neuroblastoma diagnosed in infancy by treatment era and exposures.METHODSAmong 5-year survivors of neuroblastoma in the Childhood Cancer Survivor Study diagnosed age < 1 year between 1970 and 1999, we examined the cumulative incidence of late (> 5 years from diagnosis) mortality, SMN, and CHCs (grades 2-5 and 3-5). Multivariable Cox regression models estimated hazard ratios (HRs) and 95% CIs by decade and treatment (surgery-alone v chemotherapy with or without surgery [C ± S] v radiation with or without chemotherapy ± surgery [R ± C ± S]) among survivors and between survivors and 5,051 siblings.RESULTSAmong 1,397 eligible survivors, the 25-year cumulative incidence of late mortality was 2.1% (95% CI, 1.3 to 3.9) with no difference by treatment era. Among 990 participants who completed a baseline survey, fewer survivors received radiation in more recent eras (51.2% 1970s, 20.4% 1980s, and 10.1% 1990s; P <.001). Risk of SMN was elevated only among individuals treated with radiation-containing regimens compared with surgery alone (HR[C ± S], 3.2 [95% CI, 0.9 to 11.6]; HR[R ± C ± S], 5.7 [95% CI, 1.2 to 28.1]). In adjusted models, there was a 50% reduction in risk of grade 3-5 CHCs in the 1990s versus 1970s (HR, 0.5 [95% CI, 0.3 to 0.9]; P =.01); individuals treated with radiation had a 3.6-fold risk for grade 3-5 CHCs (95% CI, 2.1 to 6.2) versus those treated with surgery alone. When compared with siblings, risk of grade 3-5 CHCs for survivors was lowest in the most recent era (HR[1970s], 4.7 [95% CI, 3.4 to 6.5]; HR[1980s], 4.6 [95% CI, 3.3 to 6.4]; HR[1990s], 2.5 [95% CI, 1.7 to 3.9]).CONCLUSIONNeuroblastoma survivors treated during infancy have a relatively low absolute burden of late mortality and SMN. Encouragingly, risk of CHCs has declined in more recent eras with reduced exposure to radiation therapy. © American Society of Clinical Oncology.
Keywords: child; retrospective studies; incidence; morbidity; retrospective study; survivor; cancer survivor; survivors; infant; neuroblastoma; neoplasms, second primary; second cancer; cancer survivors; humans; human
Journal Title: Journal of Clinical Oncology
Volume: 41
Issue: 8
ISSN: 0732-183X
Publisher: American Society of Clinical Oncology  
Date Published: 2023-03-10
Start Page: 1565
End Page: 1576
Language: English
DOI: 10.1200/jco.22.01732
PUBMED: 36525618
PROVIDER: scopus
PMCID: PMC10043581
DOI/URL:
Notes: Article -- Source: Scopus
Altmetric
Citation Impact
BMJ Impact Analytics
MSK Authors
  1. Charles A Sklar
    322 Sklar
  2. Suzanne L Wolden
    560 Wolden
  3. Danielle Novetsky Friedman
    110 Friedman