DNA copy number alterations in central primitive neuroectodermal tumors and tumors of the pineal region: an international individual patient data meta-analysis.
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DNA copy number alterations in central primitive neuroectodermal tumors and tumors of the pineal region: an international individual patient data meta-analysis. / von Bueren, André; André, O; Hagel, Christian; Hagel, Christian; Cai, Haoyang; Remke, Marc; Hasselblatt, Martin; Feuerstein, Burt G; Pernet, Sarah; Delattre, Olivier; Rutkowski, Stefan; Rutkowski, Stefan; Pfister, Stefan M; Baudis, Michael.
In: J NEURO-ONCOL, Vol. 109, No. 2, 2, 2012, p. 415-423.Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review
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TY - JOUR
T1 - DNA copy number alterations in central primitive neuroectodermal tumors and tumors of the pineal region: an international individual patient data meta-analysis.
AU - von Bueren, André
AU - André, O
AU - Hagel, Christian
AU - Hagel, Christian
AU - Cai, Haoyang
AU - Remke, Marc
AU - Hasselblatt, Martin
AU - Feuerstein, Burt G
AU - Pernet, Sarah
AU - Delattre, Olivier
AU - Rutkowski, Stefan
AU - Rutkowski, Stefan
AU - Pfister, Stefan M
AU - Baudis, Michael
PY - 2012
Y1 - 2012
N2 - Little is known about frequency, association with clinical characteristics, and prognostic impact of DNA copy number alterations (CNA) on survival in central primitive neuroectodermal tumors (CNS-PNET) and tumors of the pineal region. Searches of MEDLINE, Pubmed, and EMBASE--after the original description of comparative genomic hybridization in 1992 and July 2010--identified 15 case series of patients with CNS-PNET and tumors of the pineal region whose tumors were investigated for genome-wide CNA. One additional case study was identified from contact with experts. Individual patient data were extracted from publications or obtained from investigators, and CNAs were converted to a digitized format suitable for data mining and subgroup identification. Summary profiles for genomic imbalances were generated from case-specific data. Overall survival (OS) was estimated using the Kaplan-Meier method, and by univariable and multivariable Cox regression models. In their overall CNA profiles, low grade tumors of the pineal region clearly diverged from CNS-PNET and pineoblastoma. At a median follow-up of 89 months, 7-year OS rates of CNS-PNET, pineoblastoma, and low grade tumors of the pineal region were 22.9 ± 6, 0 ± 0, and 87.5 ± 12 %, respectively. Multivariable analysis revealed that histology (CNS-PNET), age (?2.5 years), and possibly recurrent CNAs were associated with unfavorable OS. DNA copy number profiling suggests a close relationship between CNS-PNET and pineoblastoma. Low grade tumors of the pineal region differed from CNS-PNET and pineoblastoma. Due to their high biological and clinical variability, a coordinated prospective validation in future studies is necessary to establish robust risk factors.
AB - Little is known about frequency, association with clinical characteristics, and prognostic impact of DNA copy number alterations (CNA) on survival in central primitive neuroectodermal tumors (CNS-PNET) and tumors of the pineal region. Searches of MEDLINE, Pubmed, and EMBASE--after the original description of comparative genomic hybridization in 1992 and July 2010--identified 15 case series of patients with CNS-PNET and tumors of the pineal region whose tumors were investigated for genome-wide CNA. One additional case study was identified from contact with experts. Individual patient data were extracted from publications or obtained from investigators, and CNAs were converted to a digitized format suitable for data mining and subgroup identification. Summary profiles for genomic imbalances were generated from case-specific data. Overall survival (OS) was estimated using the Kaplan-Meier method, and by univariable and multivariable Cox regression models. In their overall CNA profiles, low grade tumors of the pineal region clearly diverged from CNS-PNET and pineoblastoma. At a median follow-up of 89 months, 7-year OS rates of CNS-PNET, pineoblastoma, and low grade tumors of the pineal region were 22.9 ± 6, 0 ± 0, and 87.5 ± 12 %, respectively. Multivariable analysis revealed that histology (CNS-PNET), age (?2.5 years), and possibly recurrent CNAs were associated with unfavorable OS. DNA copy number profiling suggests a close relationship between CNS-PNET and pineoblastoma. Low grade tumors of the pineal region differed from CNS-PNET and pineoblastoma. Due to their high biological and clinical variability, a coordinated prospective validation in future studies is necessary to establish robust risk factors.
KW - Adult
KW - Humans
KW - Male
KW - Aged
KW - Female
KW - Middle Aged
KW - Adolescent
KW - Young Adult
KW - Multivariate Analysis
KW - Child
KW - Child, Preschool
KW - Infant
KW - Retrospective Studies
KW - International Cooperation
KW - Databases, Factual/statistics & numerical data
KW - Brain Neoplasms/genetics
KW - DNA Copy Number Variations/genetics
KW - Neuroectodermal Tumors, Primitive/genetics
KW - Pineal Gland/pathology
KW - Pinealoma/genetics
KW - Adult
KW - Humans
KW - Male
KW - Aged
KW - Female
KW - Middle Aged
KW - Adolescent
KW - Young Adult
KW - Multivariate Analysis
KW - Child
KW - Child, Preschool
KW - Infant
KW - Retrospective Studies
KW - International Cooperation
KW - Databases, Factual/statistics & numerical data
KW - Brain Neoplasms/genetics
KW - DNA Copy Number Variations/genetics
KW - Neuroectodermal Tumors, Primitive/genetics
KW - Pineal Gland/pathology
KW - Pinealoma/genetics
M3 - SCORING: Journal article
VL - 109
SP - 415
EP - 423
JO - J NEURO-ONCOL
JF - J NEURO-ONCOL
SN - 0167-594X
IS - 2
M1 - 2
ER -
