Abstract
BACKGROUND: Recurrent pediatric high-grade glioma (HGG) is a leading cause of cancer-related death in children. There is currently no standard of care therapy for this population. Limited survival data on pediatric patients with recurrent HGG makes design of novel clinical trials challenging. We report results of a meta-analysis investigating survival outcome in pediatric patients diagnosed with recurrent HGG over the last 20 years. METHODS: MEDLINE/PubMed, EMBASE, Web of Science and Cochrane Review databases were searched for relevant studies reporting on survival outcomes for pediatric patients with first-time recurrent HGG treated between 1996–2016. Included studies were limited to those published in English. Two independent reviewers completed study reviews. Overall survival (OS) and progression-free survival (PFS) were calculated cumulatively over all studies, by therapy subgroup, and by treatment provided pre- and post-2006. Random effects models were used for overall survival and subgroup comparisons to control for heterogeneity as measured by the I2 statistic. RESULTS: A total of 14 studies were included across 3 treatment strategies. Nine studies investigated traditional chemotherapy, 3 investigated targeted therapy, and 1 each investigated immunotherapy and radiotherapy. A total of 150 patients with a median age of 10.5 years were included. Average cumulative PFS was 55 days (95% CI 21, 88). Average cumulative OS was 201 days (95% CI 166,236). OS using targeted therapies was 215 days (95% CI 130,301) compared to 170 days (95% CI 142,198) in studies using traditional chemotherapy. OS in studies done before 2006 was 168 days (95% CI 140, 197) compared to 201 days (95% CI 166, 236) in studies completed after 2006. CONCLUSIONS: Pediatric patients with recurrent HGG suffer from poor progression-free and overall survival, regardless of therapy used. There may be a trend towards improved OS over the last 10 years and with the use of targeted therapies.