Long-term outcome of centrally located low-grade glioma in children

Cancer. 2013 Jul 15;119(14):2630-8. doi: 10.1002/cncr.28110. Epub 2013 Apr 26.

Abstract

Background: Optimal management of children with centrally located low-grade glioma (LGG) is unclear. Initial interventions in most children are chemotherapy in younger and radiation therapy (RT) in older children. A better understanding of the inherent risk factors along with the effects of interventions on long-term outcome can lead to reassessment of the current approaches to minimize long-term morbidity.

Methods: To reassess the current treatment strategies of centrally located LGG, we compared the long-term survival and morbidity of different treatment regimens. Medical records of patients primarily treated at Texas Children's Cancer and Hematology Centers between 1987 and 2008 were reviewed.

Results: Forty-seven patients with a median follow-up of 79 months were included in the analysis. The 5-year overall survival and progression-free survival (PFS) for all patients were 96% and 53%, respectively. The 5-year PFS for those treated initially with RT (12 patients; median age, 11 years [range, 3-15 years]) and with chemotherapy (28 patients; median age, 2 years [range 0-8 years]) were 76% and 37%, respectively (log-rank test P = .02). Among children who progressed after chemotherapy, the 5-year PFS after salvage RT was 55%. Patients diagnosed at a younger age (<5 years) were more likely to experience endocrine abnormalities (Fisher exact test; P<.00001).

Conclusions: Effective and durable tumor control was obtained with RT as initial treatment. In younger patients, chemotherapy can delay the use of RT; however, frequent progression and long-term morbidity are common. More effective and less toxic therapies are required in these patients, the majority of whom are long-term survivors.

Keywords: BRAF-KIAA1549 fusion protein; chemotherapy; childhood cerebral astrocytoma; childhood optic nerve glioma; hypothalamic-chiasmatic neoplasms; long-term effects; pilocytic astrocytoma; radiotherapy.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Affective Symptoms / epidemiology
  • Affective Symptoms / etiology
  • Astrocytoma / pathology
  • Astrocytoma / therapy
  • Biomarkers, Tumor / analysis*
  • Brain Neoplasms / chemistry
  • Brain Neoplasms / drug therapy
  • Brain Neoplasms / genetics
  • Brain Neoplasms / pathology*
  • Brain Neoplasms / radiotherapy
  • Brain Neoplasms / therapy*
  • Child
  • Child, Preschool
  • Cognition Disorders / epidemiology
  • Cognition Disorders / etiology
  • Disease-Free Survival
  • Endocrine System Diseases / epidemiology
  • Endocrine System Diseases / etiology
  • Female
  • Glioma / chemistry
  • Glioma / drug therapy
  • Glioma / genetics
  • Glioma / pathology*
  • Glioma / radiotherapy
  • Glioma / therapy*
  • Humans
  • In Situ Hybridization, Fluorescence
  • Infant
  • Kaplan-Meier Estimate
  • Male
  • Medical Records
  • Neoplasm Grading
  • Nervous System Diseases / epidemiology
  • Nervous System Diseases / etiology
  • Oncogene Proteins, Fusion / analysis*
  • Radiotherapy Dosage
  • Retrospective Studies
  • Survivors / statistics & numerical data
  • Texas / epidemiology
  • Treatment Outcome
  • Vision Disorders / epidemiology
  • Vision Disorders / etiology

Substances

  • BRAF-KIAA1549 fusion protein, human
  • Biomarkers, Tumor
  • Oncogene Proteins, Fusion