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Meningiomas in children and adolescents: a meta-analysis of individual patient data

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机构: [1]Princess Margaret Hosp Children, Dept Haematol & Oncol, Perth, WA 6840, Australia; [2]Princess Margaret Hosp Children, Dept Clin Res & Educ, Perth, WA 6840, Australia; [3]Univ Western Australia, Telethon Inst Child Hlth Res, Ctr Child Hlth Res, Perth, WA 6009, Australia; [4]Univ Western Australia, Sch Paediat & Child Hlth, Perth, WA 6009, Australia; [5]Univ Spital Zurich, Inst Neuropathol, Zurich, Switzerland; [6]Univ Penn, Dept Neuropathol, Childrens Hosp Philadelphia, Philadelphia, PA 19104 USA; [7]Univ Penn, Sch Med, Philadelphia, PA 19104 USA; [8]Univ Calif Davis, Med Ctr, Dept Paediat, Sect Haematol & Oncol, Sacramento, CA 95817 USA; [9]Fudan Univ, Dept Neurosurg, Huashan Hosp, Shanghai Med Coll, Shanghai 200433, Peoples R China; [10]Capital Med Univ, Beijing Tiantan Hosp, Dept Neurosurg, Beijing, Peoples R China; [11]Univ Pittsburgh, Sch Med, Dept Neurol Surg, Childrens Hosp Pittsburgh, Pittsburgh, PA 15261 USA; [12]Univ Tehran Med Sci, Dept Neurosurg, Sina Hosp, Tehran, Iran; [13]Seoul Natl Univ, Coll Med, Div Paediat Neurosurg, Seoul Natl Univ Childrens Hosp, Seoul, South Korea; [14]Natl Canc Inst, Dept Neurosurg, Rio De Janeiro, Brazil; [15]Chang Gung Univ, Coll Med, Chang Gung Childrens Hosp, Dept Paediat Neurol,Chang Gung Mem Hosp, Tao Yuan, Taiwan; [16]Hop Specialites, Dept Neurosurg, Rabat, Morocco; [17]Bombay Hosp Inst Med Sci, Dept Neurosurg, Bombay, Maharashtra, India; [18]Shandong Univ, Dept Neurosurg, Qilu Hosp, Jinan 250100, Peoples R China; [19]Natl Inst Mental Hlth & Neurosci, Dept Neurosurg, Bangalore 560029, Karnataka, India; [20]Sree Chitra Tirunal Inst Med Sci & Technol, Dept Neurosurg, Trivandrum, Kerala, India; [21]Univ Bergen, Haukeland Univ Hosp, Dept Neurosurg, Bergen, Norway; [22]Univ Bergen, Inst Surg Sci, Bergen, Norway; [23]Rigshosp, Univ Clin Neurosurg, Ctr Neurosci, DK-2100 Copenhagen, Capital Region, Denmark; [24]Princess Margaret Hosp Children, Dept Haematol & Oncol, GPO Box D184, Perth, WA 6840, Australia
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Background The epidemiological, prognostic, and therapeutic features of child and adolescent meningioma are poorly defined. Clinical knowledge has been drawn from small case series and extrapolation from adult studies. This study was done to pool and analyse the clinical evidence on child and adolescent meningioma. Methods Searches of PubMed, Medline, and Embase identified 35 case series of child and adolescent meningioma completed over the past 21 years. Individual patient data were obtained from 30 studies via direct communication with investigators. Primary outcomes were relapse-free survival (RFS) and overall survival. Prognostic variables were extent of initial surgery, use of upfront radiotherapy, age, sex, presence of neuro fibromatosis, tumour location, and tumour grade. RFS and overall survival were analysed using Kaplan-Meier survival curves and multivariable Cox regression models. Findings From a total of 677 children and adolescents with meningioma, 518 were eligible for RFS analysis and 547 for overall survival analysis. Multivariable analysis showed that patients who underwent initial gross-total resection had better RFS (hazard ratio 0.16, 95% CI 0.10-0.25; p<0.0001) and overall survival (0.21, 0.11-0.39; p<0.0001) than those who had subtotal resection. No significant benefit was seen for upfront radiotherapy in terms of RFS (0.59, 0.30-1.16; p=0.128) or overall survival (1.10, 0.53-2.28; p=0.791). Patients with neurofibromatosis type 2 (NF2) had worse RFS than those without neurofibromatosis (2.36, 1.23-4.51; p=0.010). There was a significant change in overall survival with time between patients with NF2 compared with those without neurofibromatosis (1.45, 1.09-1.92; p=0.011); although overall survival was initially better for patients with NF2 than for those without neuro fibromatosis, overall survival at 10 years was worse for patients with NF2. Patients with WHO grade III tumours had worse RFS than those with WHO grade I (3.90, 2.10-7.26; p<0.0001) and grade II tumours (2.49, 1.11-5.56; p=0.027). Interpretation Extent of initial surgical resection is the strongest independent prognostic factor for child and adolescent meningioma. No benefit for upfront radiotherapy was noted. Hence, aggressive surgical management, to achieve gross-total resection, is the initial treatment of choice. In the event of a subtotal resection, repeat resection is recommended to achieve maximum extirpation. Close observation is warranted for patients who have a subtotal resection or who have WHO grade III tumours. Patients without neuro fibromatosis should have a minimum 10-year follow-up, whereas patients with NF2 should be considered a special risk category, necessitating life-long follow-up.

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出版当年[2010]版:
大类 | 1 区 医学
小类 | 1 区 肿瘤学
最新[2023]版:
大类 | 1 区 医学
小类 | 1 区 肿瘤学
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出版当年[2009]版:
Q1 ONCOLOGY
最新[2023]版:
Q1 ONCOLOGY

影响因子: 最新[2023版] 最新五年平均 出版当年[2009版] 出版当年五年平均 出版前一年[2008版] 出版后一年[2010版]

第一作者:
第一作者机构: [1]Princess Margaret Hosp Children, Dept Haematol & Oncol, Perth, WA 6840, Australia; [3]Univ Western Australia, Telethon Inst Child Hlth Res, Ctr Child Hlth Res, Perth, WA 6009, Australia; [4]Univ Western Australia, Sch Paediat & Child Hlth, Perth, WA 6009, Australia;
通讯作者:
通讯机构: [1]Princess Margaret Hosp Children, Dept Haematol & Oncol, Perth, WA 6840, Australia; [3]Univ Western Australia, Telethon Inst Child Hlth Res, Ctr Child Hlth Res, Perth, WA 6009, Australia; [4]Univ Western Australia, Sch Paediat & Child Hlth, Perth, WA 6009, Australia; [24]Princess Margaret Hosp Children, Dept Haematol & Oncol, GPO Box D184, Perth, WA 6840, Australia
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