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• W2896063423 abstract "Atypical teratoid/rhabdoid tumor (AT/RT) is a rare and highly aggressive pediatric CNS malignant tumor mainly affects infants and young children. The purpose of the study is to evaluate the possible prognostic factors and optimal management for this rare disease. Twenty-eight pediatric patients of CNS AT/RT treated with radiation therapy (RT) as part of multimodality treatment at one institution between 1996 and 2015 were retrospectively reviewed. Progression free survival (PFS), overall survival (OS), and neuro-axis failure free survival were analyzed in correlation with possible prognostic factors. The median age at diagnosis was 4.3 years old (range 0.6-19 years), with 12 patients (42.9%) less than 3 years old. Six patients (21.4%) had neuro-axis spreading at diagnosis. Radiotherapy was delayed for a longer period after surgery in patients less than 3 years old compared to older ones, with mean interval 4.9 and 1.9 months, respectively (p=0.06). Chemotherapy was given before RT initiation in 14 patients (50.0%). All of the patients ≥ 3 years old received cranio-spinal irradiation (CSI) followed by boost to tumor bed, while only 4 patients < 3 years-old(33.3%) did so (p<0.01). Mean dose to the primary tumor bed was 48.7 Gy (range 30.0-56.0 Gy), and mean CSI dose was 29.8Gy (range 24.0-36.0 Gy). Seven of 12 patients < 3 years old received high dose chemotherapy and autologous peripheral blood stem cell (PBSC) rescue. The median follow-up period was 45 months. The median PFS was 11 months and OS was 67 months. Three-year PFS was 46.4%, and OS was 60.7%. At the end of follow-up, 15 patients (53.6%) suffered from disease relapse; 13 of them had disseminating neuraxis seeding, and 11 died within one year after relapse. Interval between surgery and RT > 2 months (HR= 3.38, p=0.01) was correlated with worse PFS, while higher tumor bed RT dose (HR 0.9, p<0.01) was associated with better PFS. Overall survival was significantly worse with interval between surgery and RT > 2 months (HR=3.54, p=0.02). Cranio-spinal irradiation increased neuraxis failure free survival (HR 0.36, p= 0.05). In the group of patients < 3 years old, high dose chemotherapy followed by autologous PBSC rescue was associated with better OS compared to intravenous +/- intra-thecal chemotherapy alone (HR 0.15, p=0.04). Early and aggressive RT immediately after surgery is critical for disease control. Prognosis is extremely poor after relapse. For younger patients, high dose chemotherapy with autologous PBSC rescue provides better outcome, and may be considered as an alternative treatment to cranio-spinal irradiation." @default.
• W2896063423 created "2018-10-26" @default.
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• W2896063423 date "2018-11-01" @default.
• W2896063423 modified "2023-10-14" @default.
• W2896063423 title "Pediatric Central Nervous System Atypical Teratoid/Rhabdoid Tumor: Early and Aggressive Post-Operative Radiation Therapy Provides Better Outcome" @default.
• W2896063423 doi "https://doi.org/10.1016/j.ijrobp.2018.07.1344" @default.
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