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MENINGIOMAS IN CHILDREN AND ADOLESCENTS: A META-ANALYSIS OF INDIVIDUAL PATIENT DATA
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Affilliation
Princess Margaret Hospital for Children. Department of Haematology and Oncology. Perth, WA, Australia / University of Western Australia. Centre for Child Health Research. Perth, WA, Australia / Telethon Institute for Child Health Research. Perth, WA, Australia / University of Western Australia. School of Paediatrics and Child Health. Perth, WA, Australia.
Princess Margaret Hospital for Children. Department of Clinical Research and Education. Perth, WA, Australia / University of Western Australia. School of Paediatrics and Child Health. Perth, WA, Australia.
UniversitätsSpital Zürich. Institut für Neuropathologie. Zürich, Switzerland.
University of Pennsylvania. School of Medicine. Department of Neuropathology. Philadelphia, PA, USA / The Children's Hospital of Philadelphia. Philadelphia, PA, USA.
University of California. Davis Medical Centre. Department of Paediatrics. Section Haematology and Oncology. Sacramento, CA, USA.
Fudan University. Shanghai Medical College. Department of Neurosurgery. Shanghai, China / Huashan Hospital. Shanghai, China.
Capital Medical University. Beijing Tiantan Hospital. Department of Neurosurgery. Beijing, China.
University of Pittsburgh. Medical School. Children's Hospital of Pittsburgh. Department of Neurological Surgery. Pittsburgh, PA, USA
Tehran University of Medical Sciences. Sina Hospital. Department of Neurosurgery. Tehran, Iran.
Seoul National University. College of Medicine. Seoul National University Children's Hospital. Division of Paediatric Neurosurgery. Seoul, Korea.
National Cancer Institute. Department of Neurosurgery. Rio de Janeiro, RJ, Brazil.
Chang Gung University. College of Medicine. Chang Gung Memorial Hospital. Department of Paediatric Neurology. Chang Gung Children's Hospital. Taoyuan, Taiwan.
Hôpital des Spécialités.Department of Neurosurgery. Rabat, Morocco.
Bombay Hospital Institute of Medical Sciences. Department of Neurosurgery. Mumbai, India.
Qilu Hospital of Shandong University. Department of Neurosurgery. Jinan, China.
National Institute of Mental Health and Neurosciences. Department of Neurosurgery. Bangalore, India
Sree Chitra Tirunal Institute for Medical Sciences and Technology. Department of Neurosurgery. Trivandrum, India.
Department of Neurosurgery, Haukeland University Hospital, and Institute of Surgical Science, University of Bergen, Bergen, Norway
University Clinic of Neurosurgery. Rigshospitalet. Neuroscience Centre. Copenhagen, Denmark.
Princess Margaret Hospital for Children. Department of Haematology and Oncology. Perth, WA, Australia / University of Western Australia. School of Paediatrics and Child Health. Perth, WA, Australia.
Princess Margaret Hospital for Children. Department of Haematology and Oncology. Perth, WA, Australia / University of Western Australia. Centre for Child Health Research. Perth, WA, Australia / Telethon Institute for Child Health Research. Perth, WA, Australia / University of Western Australia. School of Paediatrics and Child Health. Perth, WA, Australia.
Princess Margaret Hospital for Children. Department of Clinical Research and Education. Perth, WA, Australia / University of Western Australia. School of Paediatrics and Child Health. Perth, WA, Australia.
UniversitätsSpital Zürich. Institut für Neuropathologie. Zürich, Switzerland.
University of Pennsylvania. School of Medicine. Department of Neuropathology. Philadelphia, PA, USA / The Children's Hospital of Philadelphia. Philadelphia, PA, USA.
University of California. Davis Medical Centre. Department of Paediatrics. Section Haematology and Oncology. Sacramento, CA, USA.
Fudan University. Shanghai Medical College. Department of Neurosurgery. Shanghai, China / Huashan Hospital. Shanghai, China.
Capital Medical University. Beijing Tiantan Hospital. Department of Neurosurgery. Beijing, China.
University of Pittsburgh. Medical School. Children's Hospital of Pittsburgh. Department of Neurological Surgery. Pittsburgh, PA, USA
Tehran University of Medical Sciences. Sina Hospital. Department of Neurosurgery. Tehran, Iran.
Seoul National University. College of Medicine. Seoul National University Children's Hospital. Division of Paediatric Neurosurgery. Seoul, Korea.
National Cancer Institute. Department of Neurosurgery. Rio de Janeiro, RJ, Brazil.
Chang Gung University. College of Medicine. Chang Gung Memorial Hospital. Department of Paediatric Neurology. Chang Gung Children's Hospital. Taoyuan, Taiwan.
Hôpital des Spécialités.Department of Neurosurgery. Rabat, Morocco.
Bombay Hospital Institute of Medical Sciences. Department of Neurosurgery. Mumbai, India.
Qilu Hospital of Shandong University. Department of Neurosurgery. Jinan, China.
National Institute of Mental Health and Neurosciences. Department of Neurosurgery. Bangalore, India
Sree Chitra Tirunal Institute for Medical Sciences and Technology. Department of Neurosurgery. Trivandrum, India.
Department of Neurosurgery, Haukeland University Hospital, and Institute of Surgical Science, University of Bergen, Bergen, Norway
University Clinic of Neurosurgery. Rigshospitalet. Neuroscience Centre. Copenhagen, Denmark.
Princess Margaret Hospital for Children. Department of Haematology and Oncology. Perth, WA, Australia / University of Western Australia. School of Paediatrics and Child Health. Perth, WA, Australia.
Princess Margaret Hospital for Children. Department of Haematology and Oncology. Perth, WA, Australia / University of Western Australia. Centre for Child Health Research. Perth, WA, Australia / Telethon Institute for Child Health Research. Perth, WA, Australia / University of Western Australia. School of Paediatrics and Child Health. Perth, WA, Australia.
Abstract
Background: The epidemiological, prognostic, and therapeutic features of child and adolescent meningioma are poorly defi ned. 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 identifi ed 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 neurofibromatosis, 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 signifi cant benefi t 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 neurofi bromatosis type 2 (NF2) had worse RFS than those without neurofi bromatosis (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 neurof bromatosis (1·45, 1·09–1·92; p=0·011); although overall survival was initially better for patients with NF2 than for those without neurof bromatosis, 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 fi bromatosis 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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