Lennox Byer1, Cassie N Kline2,3, Christina Coleman4, Isabel E Allen5, Evans Whitaker6, Sabine Mueller7,8,9,10. 1. School of Medicine, University of California, San Francisco, 513 Parnassus Avenue, San Francisco, CA, 94143, USA. 2. Division of Hematology/Oncology, Department of Pediatrics, University of California, San Francisco, 550 16th Street, 4th Floor, San Francisco, CA, 94158, USA. 3. Department of Neurology, University of California, San Francisco, Box 0434, 625 Nelson Rising Lane, San Francisco, CA, 94158, USA. 4. Department of Pediatrics, Division of Pediatric Hematology/Oncology, UCSF Benioff Children's Hospital, Oakland, 747 52nd Street, Oakland, CA, 94609, USA. 5. Department of Epidemiology & Biostatistics, University of California, San Francisco, 550 16th Street, 2nd Floor, San Francisco, CA, 94158, USA. 6. UCSF Health Sciences Library, University of California, San Francisco, 530 Parnassus Ave, San Francisco, CA, 94143, USA. 7. Department of Neurology, University of California, San Francisco, Box 0434, 625 Nelson Rising Lane, San Francisco, CA, 94158, USA. sabine.mueller@ucsf.edu. 8. Department of Pediatrics, Division of Pediatric Hematology/Oncology, UCSF Benioff Children's Hospital, Oakland, 747 52nd Street, Oakland, CA, 94609, USA. sabine.mueller@ucsf.edu. 9. Department of Neurological Surgery, University of California, San Francisco, 505 Parnassus Avenue, M779, San Francisco, CA, USA. sabine.mueller@ucsf.edu. 10. University Children's Hospital Zuerich, Zurich, Switzerland. sabine.mueller@ucsf.edu.
Abstract
PURPOSE: The purpose of this study was to determine outcomes in recurrent pediatric ependymoma. METHODS: We performed a systematic review of PubMed, Embase, Web of Science and the Cochrane Library for studies reporting on survival outcomes for pediatric patients with recurrent ependymoma. We then performed a meta-analysis of all eligible results. Survival outcomes were identified across location of recurrence, therapy at recurrence, and age at recurrence. RESULTS: Eleven studies met final inclusion criteria. Pooled median progression free survival (PFS) from date of first recurrence was 6.7 months (95% confidence interval [95% CI] 4.7-8.8). Pooled median overall survival (OS) from date of first recurrence was 11.2 months (95% CI 6.4-16.0). Participants with supratentorial recurrences demonstrated a shorter OS of 8.3 months (95% CI 3.2-13.3) compared to 20.1 months (95% CI 8.4-31.7) for those with infratentorial recurrence. Patients who underwent surgery at recurrence had a median OS of 24.2 months (95% CI 14.2-34.1) compared to 29.2 months (95% CI 17.4-41.1) in those who received radiation compared to 19.3 months (95% CI 10.3-28.3) in those who received chemotherapy. Patients younger than age 3 years at time of recurrence demonstrated a median OS of 31.0 months (95% CI - 25.3-87.3) compared to 17.5 months (95% CI 9.9-25.2) for those that recurred beyond 3 years of age. CONCLUSIONS: Our findings illustrate that children with recurrent ependymoma suffer from poor outcomes; however, these outcomes range widely depending on patient, tumor, and treatment characteristics. New therapies and treatment strategies are needed to improve outcomes in this group.
PURPOSE: The purpose of this study was to determine outcomes in recurrent pediatric ependymoma. METHODS: We performed a systematic review of PubMed, Embase, Web of Science and the Cochrane Library for studies reporting on survival outcomes for pediatric patients with recurrent ependymoma. We then performed a meta-analysis of all eligible results. Survival outcomes were identified across location of recurrence, therapy at recurrence, and age at recurrence. RESULTS: Eleven studies met final inclusion criteria. Pooled median progression free survival (PFS) from date of first recurrence was 6.7 months (95% confidence interval [95% CI] 4.7-8.8). Pooled median overall survival (OS) from date of first recurrence was 11.2 months (95% CI 6.4-16.0). Participants with supratentorial recurrences demonstrated a shorter OS of 8.3 months (95% CI 3.2-13.3) compared to 20.1 months (95% CI 8.4-31.7) for those with infratentorial recurrence. Patients who underwent surgery at recurrence had a median OS of 24.2 months (95% CI 14.2-34.1) compared to 29.2 months (95% CI 17.4-41.1) in those who received radiation compared to 19.3 months (95% CI 10.3-28.3) in those who received chemotherapy. Patients younger than age 3 years at time of recurrence demonstrated a median OS of 31.0 months (95% CI - 25.3-87.3) compared to 17.5 months (95% CI 9.9-25.2) for those that recurred beyond 3 years of age. CONCLUSIONS: Our findings illustrate that children with recurrent ependymoma suffer from poor outcomes; however, these outcomes range widely depending on patient, tumor, and treatment characteristics. New therapies and treatment strategies are needed to improve outcomes in this group.
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