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Gliomatosis cerebri in children: A poor prognostic phenotype of diffuse gliomas with a distinct molecular profile
G. Nussbaumer, M. Benesch, Y. Grabovska, A. Mackay, D. Castel, J. Grill, MM. Alonso, M. Antonelli, S. Bailey, JN. Baugh, V. Biassoni, M. Blattner-Johnson, A. Broniscer, A. Carai, GS. Colafati, N. Colditz, S. Corbacioglu, S. Crampsie, N....
Jazyk angličtina Země Anglie, Velká Británie
Typ dokumentu časopisecké články, multicentrická studie
Grantová podpora
German- and Styrian Childhood Cancer Foundation
Etoile de Martin
Les Boucles du Cœur de la Fondation Carrefour
BB-033-00065
Necker Imagine Tumor and DNA biobank
Rudy A Menon Foundation
CRIS Cancer Foundation
Ollie Young Foundation
C13468/A23536
Cancer Research UK - United Kingdom
Joshua Bembo Project and the AYJ Fund
NHS
NIHR Great Ormond Street Hospital Biomedical Research Centre
PRIMUS/19/MED/06
Izas, la Princesa Guisante Foundation
Charles University Grant Agency
Fondazione Heal
Il Coraggio dei Bambini
Martina e La Sua Luna
Il laboratorio di Chiara
NET-2019-12371188
Regione Lombardia
Italian Ministry of Health
NET-2019-12371188
Regione Liguria
NLK
Free Medical Journals
od 1999 do Před 1 rokem
PubMed Central
od 1999 do Před 1 rokem
Europe PubMed Central
od 1999 do Před 1 rokem
Open Access Digital Library
od 1999-01-01
Medline Complete (EBSCOhost)
od 2004-01-01 do Před 1 rokem
PubMed
38717379
DOI
10.1093/neuonc/noae080
Knihovny.cz E-zdroje
- MeSH
- dítě MeSH
- fenotyp MeSH
- gliom * genetika patologie MeSH
- kojenec MeSH
- lidé MeSH
- metylace DNA MeSH
- míra přežití MeSH
- mladiství MeSH
- mutace MeSH
- nádorové biomarkery genetika MeSH
- nádory mozku * genetika patologie MeSH
- následné studie MeSH
- neuroepitelové nádory * patologie genetika MeSH
- předškolní dítě MeSH
- prognóza MeSH
- retrospektivní studie MeSH
- stupeň nádoru MeSH
- Check Tag
- dítě MeSH
- kojenec MeSH
- lidé MeSH
- mladiství MeSH
- mužské pohlaví MeSH
- předškolní dítě MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- multicentrická studie MeSH
BACKGROUND: The term gliomatosis cerebri (GC), a radiology-defined highly infiltrating diffuse glioma, has been abandoned since molecular GC-associated features could not be established. METHODS: We conducted a multinational retrospective study of 104 children and adolescents with GC providing comprehensive clinical and (epi-)genetic characterization. RESULTS: Median overall survival (OS) was 15.5 months (interquartile range, 10.9-27.7) with a 2-year survival rate of 28%. Histopathological grading correlated significantly with median OS: CNS WHO grade II: 47.8 months (25.2-55.7); grade III: 15.9 months (11.4-26.3); grade IV: 10.4 months (8.8-14.4). By DNA methylation profiling (n = 49), most tumors were classified as pediatric-type diffuse high-grade glioma (pedHGG), H3-/IDH-wild-type (n = 31/49, 63.3%) with enriched subclasses pedHGG_RTK2 (n = 19), pedHGG_A/B (n = 6), and pedHGG_MYCN (n = 5), but only one pedHGG_RTK1 case. Within the pedHGG, H3-/IDH-wild-type subgroup, recurrent alterations in EGFR (n = 10) and BCOR (n = 9) were identified. Additionally, we observed structural aberrations in chromosome 6 in 16/49 tumors (32.7%) across tumor types. In the pedHGG, H3-/IDH-wild-type subgroup TP53 alterations had a significant negative effect on OS. CONCLUSIONS: Contrary to previous studies, our representative pediatric GC study provides evidence that GC has a strong predilection to arise on the background of specific molecular features (especially pedHGG_RTK2, pedHGG_A/B, EGFR and BCOR mutations, chromosome 6 rearrangements).
2nd Department of Pediatrics Semmelweis University Budapest Hungary
AbbVie Inc Oncology Development North Chicago IL U S A
Department of Neurosciences University of Turin Turin Italy
Department of Onco Hematology Gene and Cell Therapy Bambino Gesù Children's Hospital Rome Italy
Department of Pathology Amsterdam UMC Amsterdam The Netherlands
Department of Pediatric Hematology and Oncology University Hospitals Leuven Belgium
Department of Pediatric Oncology and Hematology Saarland University Homburg Germany
Department of Pediatrics Clínica Universidad de Navarra Pamplona Spain
Department of Radiation Oncology University of Leipzig Leipzig Germany
Department of Radiology and Nuclear Medicine Erasmus MC Rotterdam The Netherlands
Division of Clinical Neuro Oncology Department of Neurology University Hospital Bonn Bonn Germany
Division of Hematology Oncology Children's Hospital of Pittsburgh Pittsburgh PA U S A
Division of Molecular Pathology Institute of Cancer Research London UK
Division of Pediatric Glioma Research German Cancer Research Center Heidelberg Germany
Division of Pediatric Hematology and Oncology University Medical Center Göttingen Göttingen Germany
Division of Pediatric Hematology Oncology Institute of Oncology Istanbul University Istanbul Turkey
Division of Pediatric Neurooncology German Cancer Research Center Heidelberg Germany
GHU Paris Psychiatry and Neuroscience Sainte Anne Hospital Department of Neuropathology Paris France
Health Research Institute of Navarra Pamplona Navarra Spain
Hopp Children's Cancer Center Heidelberg Germany
Institute of Biostatistics and Clinical Research University of Münster Münster Germany
Institute of Diagnostic and Interventional Neuroradiology University of Würzburg Würzburg Germany
National Center for Tumor Diseases Heidelberg Heidelberg Germany
Neuro Oncology Unit IRCSS Istituto Giannina Gaslini Genoa Italy
Neurosurgery Unit Bambino Gesù Children's Hospital Rome Italy
Oncological Neuroradiology and Advanced Diagnostics Unit Bambino Gesù Children's Hospital Rome Italy
Pathology Unit Bambino Gesù Children's Hospital Rome Italy
Pediatric Neuro Oncology Pediatric Cancer Center Barcelona Hospital Sant Joan de Deu Barcelona Spain
Pediatric Oncology Department of Oncology KU Leuven Belgium
Pediatric Oncology Department University Hospital São João Porto Portugal
Pediatric Oncology Great Ormond Street Hospital for Children London UK
Pediatric Oncology Unit Fondazione IRCCS Istituto Nazionale dei Tumori Milano Italy
Princess Máxima Center for Pediatric Oncology Utrecht The Netherlands
Solid Tumor Program Center for the Applied Medical Research Pamplona Navarra Spain
Citace poskytuje Crossref.org
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