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


Authors: Wells, E. M.; Ullrich, N. J.; Seidel, K.; Leisenring, W.; Sklar, C. A.; Armstrong, G. T.; Diller, L.; King, A.; Krull, K. R.; Neglia, J. P.; Stovall, M.; Whelan, K.; Oeffinger, K. C.; Robison, L. L.; Packer, R. J.
Article Title: Longitudinal assessment of late-onset neurologic conditions in survivors of childhood central nervous system tumors: A Childhood Cancer Survivor Study report
Abstract: 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. © 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.
Keywords: adolescent; adult; child; controlled study; major clinical study; radiation dose; incidence; cohort analysis; risk factor; central nervous system tumor; childhood cancer; late effects; risk assessment; dizziness; tumor recurrence; cataract; seizure; headache; meningioma; glaucoma; sibling; motor dysfunction; neurologic disease; hearing impairment; cerebrovascular accident; longitudinal study; vertigo; coordination disorder; childhood cancer survivor; human; male; female; article; neurologic outcomes; childhood central nervous system tumor survivors; late onset disorder
Journal Title: Neuro-Oncology
Volume: 20
Issue: 1
ISSN: 1522-8517
Publisher: Oxford University Press  
Date Published: 2018-01-01
Start Page: 132
End Page: 142
Language: English
DOI: 10.1093/neuonc/nox148
PROVIDER: scopus
PMCID: PMC5761581
PUBMED: 29016809
DOI/URL:
Notes: Article -- Export Date: 1 March 2018 -- Source: Scopus
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  1. Charles A Sklar
    322 Sklar