Longitudinal assessment of late-onset neurologic conditions in survivors of childhood central nervous system tumors: A Childhood Cancer Survivor Study report

Elizabeth M. Wells, Nicole J. Ullrich, Kristy Seidel, Wendy Leisenring, Charles A. Sklar, Gregory T. Armstrong, Lisa DIller, Allison King, Kevin R. Krull, Joseph P. Neglia, Marilyn Stovall, Kimberly Whelan, Kevin C. Oeffinger, Leslie L. Robison, Roger J. Packer

Research output: Contribution to journalArticlepeer-review

36 Scopus citations


Background Survivors of childhood central nervous system (CNS) tumors experience high rates of treatment-related neurologic sequelae. Whether survivors continue to be at increased risk for new events as they age is unknown. Methods Adverse neurologic health conditions in 5-year survivors of CNS tumors from the Childhood Cancer Survivor Study (n = 1876) were evaluated longitudinally at a median 23.0 years from diagnosis (range, 5.1-38.9), median age at last evaluation 30.3 years (range, 6.1-56.4). Multivariable regression estimated hazard ratios (HRs) and 95% CIs. Results From 5 to 30 years post diagnosis, cumulative incidence increased for seizures from 27% to 41%, motor impairment 21% to 35%, and hearing loss 9% to 23%. Risks were elevated compared with siblings (eg, seizures HR: 12.7; 95% CI: 9.6-16.7; motor impairment HR: 7.6; 95% CI: 5.8-9.9; hearing loss HR: 18.4; 95% CI: 13.1-25.9). Regional brain doses of radiation therapy were associated with development of new deficits (eg, frontal ≥50 Gy and motor impairment HR: 2.0; 95% CI: 1.2-3.4). Increased risk for motor impairment was also associated with tumor recurrence (HR: 2.6; 95% CI: 1.8-3.8), development of a meningioma (HR: 2.3; 95% CI: 0.9-5.4), and stroke (HR: 14.9; 95% CI: 10.4-21.4). Seizure risk was doubled by recurrence (HR: 2.3; 95% CI: 1.6-3.2), meningioma (HR: 2.6; 95% CI: 1.1-6.5), and stroke (HR: 2.0; 95% CI: 1.1-3.4). Conclusions CNS tumor survivors remain at risk for new-onset adverse neurologic events across their lifespans at a rate greater than siblings. Cranial radiation, stroke, tumor recurrence, and development of meningioma were independently associated with late-onset adverse neurologic sequelae.

Original languageEnglish (US)
Pages (from-to)132-142
Number of pages11
Issue number1
StatePublished - Jan 1 2018

Bibliographical note

Funding Information:
This work was supported by the National Cancer Institute (CA55727, G.  T. Armstrong, Principal Investigator). Support to St Jude Children’s Research Hospital was also provided by the Cancer Center Support (CORE) grant (CA21765, C. Roberts, Principal Investigator) and the American Lebanese-Syrian Associated Charities (ALSAC). Investigators interested in potential uses of this resource are encouraged to visit: http://ccss.stjude.org. E.M.W.  was supported by

Publisher Copyright:
© The Author(s) 2017. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.


  • childhood central nervous system tumor survivors
  • late effects
  • neurologic outcomes


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