Childhood Atypical Teratoid Rhabdoid Tumor of the Central Nervous System A Meta-Analysis of Observational Studies

被引:121
作者
Athale, Uma H. [1 ,2 ]
Duckworth, JoAnn [2 ]
Odame, Isaac [3 ]
Barr, Ronald [2 ]
机构
[1] McMaster Univ, Hlth Sci Ctr, Div Hematol Oncol, Dept Pediat, Hamilton, ON L8N 3Z5, Canada
[2] McMaster Childrens Hosp, Hamilton Hlth Sci Corp, Hematol Oncol Serv, Hamilton, ON, Canada
[3] Hosp Sick Children, Div Hematol, Toronto, ON M5G 1X8, Canada
关键词
atypical teratoid rhabdoid tumor; central nervous system; intrathecal chemotherapy; radiation therapy; children; LONG-TERM SURVIVAL; TERATOID/RHABDOID TUMOR; THE-LITERATURE; RADIATION-THERAPY; PATHOLOGICAL FEATURES; MR FINDINGS; BRAIN; INFANCY; CHILDREN; CNS;
D O I
10.1097/MPH.0b013e3181b258a9
中图分类号
R73 [肿瘤学];
学科分类号
100214 ;
摘要
Purpose: Therapy for central nervous system (CNS) atypical teratoid rhabdoid tumor (ATRT) is controversial. We describe 4 children treated with sarcoma-like therapy and review the literature to evaluate outcome in relation to treatment modalities. Procedure: Reports from 1995 to 2007, describing clinical features of children (:5 18 years) were reviewed for details of demography, therapy, and outcome. Kaplan-Meier survival analyses were used to study the impact of clinical features, demography, and therapy on overall survival (OS). Results: The median OS for patients treated with multiagent chemotherapy (n = 79) was 17.3 months (range, 1.5-93 mo); unrelated to age at diagnosis, sex, tumor site, and extent of resection. Overall mortality was 67%. Disease progression (craniospinal spread in 58%) was the major cause of death. Patients (n = 30) treated with intrathecal (IT) chemotherapy had significantly higher 2-year. OS [64% (95% confidence interval, 46.5-82.0) vs. 17.3% (95% confidence interval, 5.4-29.3); P < 0.0001] and lower prevalence of distant CNS metastasis compared with those without IT therapy (n = 49) (20% vs. 59.2%; P = 0.001). Conclusions: Despite dismal OS, multimodal therapy can induce remission even in metastatic CNS ATRT with partial resection. IT chemotherapy results in higher OS and, because of an overall high rate of distant relapse, should be considered in future trials.
引用
收藏
页码:651 / 663
页数:13
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