title: Survival and long-term health and cognitive outcomes after low-grade glioma.
abstract: Long-term morbidity for children with low-grade glioma (LGG) requires exposure-specific characterization. Overall survival (OS) and progression-free survival (PFS) were estimated for 361 children diagnosed with LGG between 1985 and 2007 at a single institution. Five-year survivors (n = 240) received risk-based clinical assessment. Cumulative incidence of late effects 15 years from diagnosis were estimated. Risk factors for adverse health were identified using Fine and Gray's approach to Cox's proportional hazards model, accounting for death as a competing risk. OS at 15 years was 86% (95% confidence interval [CI] 82%-90%), and PFS was 55% (95% CI 51%-58%). Among the 240 5-year survivors, the 5-, 10-, and 15-year cumulative incidence of adverse outcomes included blindness: 10%, 13%, and 18%, respectively; hearing loss: 8%, 14%, and 22%; obesity/overweight: 18%, 35%, and 53%; hyperinsulinism: 1%, 5%, and 24%; growth hormone deficiency: 13%, 27%, and 29%;thyroid hormone deficiency: 16%, 28%, and 33%; and adrenocorticotropic hormone (ACTH) deficiency: 12%, 22%, and 26%. Multivariable models demonstrated radiation therapy to be a significant independent predictor of hearing loss, growth hormone deficiency, abnormal thyroid function, and ACTH deficiency. Diencephalic location was a statistically significant independent risk factor for blindness, growth hormone deficiency, abnormal thyroid function, and ACTH deficiency. Among the 182 5-year survivors assessed for intellectual function, 34% had an intelligence quotient (IQ) below average (<85), associated with younger age at diagnosis, epilepsy, and shunt placement. Survivors of childhood LGG experience substantial long-term adverse effects that continue to increase well beyond the 5-year survival time point.
authors.affiliation: Department of Epidemiology & Cancer Control, St. Jude Children's Research Hospital, 262 Danny Thomas Place, Mail Stop 735, Memphis, TN 38105, USA. greg.armstrong@stjude.org
authors.forename: Gregory T
authors.initials: GT
authors.lastname: Armstrong
authors.forename: Heather M
authors.initials: HM
authors.lastname: Conklin
authors.forename: Sujuan
authors.initials: S
authors.lastname: Huang
authors.forename: Deokumar
authors.initials: D
authors.lastname: Srivastava
authors.forename: Robert
authors.initials: R
authors.lastname: Sanford
authors.forename: David W
authors.initials: DW
authors.lastname: Ellison
authors.forename: Thomas E
authors.initials: TE
authors.lastname: Merchant
authors.forename: Melissa M
authors.initials: MM
authors.lastname: Hudson
authors.forename: Mary Ellen
authors.initials: ME
authors.lastname: Hoehn
authors.forename: Leslie L
authors.initials: LL
authors.lastname: Robison
authors.forename: Amar
authors.initials: A
authors.lastname: Gajjar
authors.forename: E Brannon
authors.initials: EB
authors.lastname: Morris
mesh_terms: D000293:Adolescent; D000328:Adult; D001932:Brain Neoplasms / Q000150:complications / Q000401:mortality* / Q000628:therapy; D002648:Child; D002675:Child, Preschool; D003072:Cognition Disorders / Q000209:etiology / Q000401:mortality*; D005260:Female; D005500:Follow-Up Studies; D005910:Glioma / Q000150:complications / Q000401:mortality* / Q000628:therapy; D006304:Health Status; D006801:Humans; D015994:Incidence; D007223:Infant; D007231:Infant, Newborn; D008297:Male; D009017:Morbidity; D011379:Prognosis; D012307:Risk Factors; D015996:Survival Rate; D017741:Survivors; D055815:Young Adult