{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,14]],"date-time":"2026-08-14T17:35:38Z","timestamp":1786728938825,"version":"build-2736575974"},"reference-count":44,"publisher":"Oxford University Press (OUP)","issue":"9","license":[{"start":{"date-parts":[[2024,5,8]],"date-time":"2024-05-08T00:00:00Z","timestamp":1715126400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"German- and Styrian Childhood Cancer Foundation"},{"name":"Etoile de Martin"},{"name":"Les Boucles du C\u0153ur de la Fondation Carrefour"},{"name":"Necker Imagine Tumor and DNA biobank","award":["BB-033-00065"],"award-info":[{"award-number":["BB-033-00065"]}]},{"name":"Rudy A Menon Foundation"},{"DOI":"10.13039\/501100023479","name":"CRIS Cancer Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100023479","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Ollie Young Foundation"},{"DOI":"10.13039\/501100000289","name":"Cancer Research UK","doi-asserted-by":"publisher","award":["C13468\/A23536"],"award-info":[{"award-number":["C13468\/A23536"]}],"id":[{"id":"10.13039\/501100000289","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000289","name":"Cancer Research UK","doi-asserted-by":"publisher","award":["DRCRPG-Nov21\\100002"],"award-info":[{"award-number":["DRCRPG-Nov21\\100002"]}],"id":[{"id":"10.13039\/501100000289","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Joshua Bembo Project and the AYJ Fund"},{"DOI":"10.13039\/100030827","name":"NHS","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100030827","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100019256","name":"NIHR Great Ormond Street Hospital Biomedical Research Centre","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100019256","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Izas, la Princesa Guisante Foundation","award":["PRIMUS\/19\/MED\/06"],"award-info":[{"award-number":["PRIMUS\/19\/MED\/06"]}]},{"DOI":"10.13039\/100007543","name":"Charles University Grant Agency","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007543","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Fondazione Heal"},{"name":"Il Coraggio dei Bambini"},{"name":"Martina e La Sua Luna"},{"name":"Il laboratorio di Chiara"},{"DOI":"10.13039\/501100009882","name":"Regione Lombardia","doi-asserted-by":"publisher","award":["NET-2019-12371188"],"award-info":[{"award-number":["NET-2019-12371188"]}],"id":[{"id":"10.13039\/501100009882","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003196","name":"Italian Ministry of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003196","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100009881","name":"Regione Liguria","doi-asserted-by":"publisher","award":["NET-2019-12371188"],"award-info":[{"award-number":["NET-2019-12371188"]}],"id":[{"id":"10.13039\/501100009881","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,9,5]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>The term gliomatosis cerebri (GC), a radiology-defined highly infiltrating diffuse glioma, has been abandoned since molecular GC-associated features could not be established.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>We conducted a multinational retrospective study of 104 children and adolescents with GC providing comprehensive clinical and (epi-)genetic characterization.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Median overall survival (OS) was 15.5 months (interquartile range, 10.9\u201327.7) with a 2-year survival rate of 28%. Histopathological grading correlated significantly with median OS: CNS WHO grade II: 47.8 months (25.2\u201355.7); grade III: 15.9 months (11.4\u201326.3); grade IV: 10.4 months (8.8\u201314.4). By DNA methylation profiling (n\u2005=\u200549), most tumors were classified as pediatric-type diffuse high-grade glioma (pedHGG), H3-\/IDH-wild-type (n\u2005=\u200531\/49, 63.3%) with enriched subclasses pedHGG_RTK2 (n\u2005=\u200519), pedHGG_A\/B (n\u2005=\u20056), and pedHGG_MYCN (n\u2005=\u20055), but only one pedHGG_RTK1 case. Within the pedHGG, H3-\/IDH-wild-type subgroup, recurrent alterations in EGFR (n\u2005=\u200510) and BCOR (n\u2005=\u20059) 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.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>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).<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/neuonc\/noae080","type":"journal-article","created":{"date-parts":[[2024,5,8]],"date-time":"2024-05-08T14:30:25Z","timestamp":1715178625000},"page":"1723-1737","source":"Crossref","is-referenced-by-count":13,"title":["Gliomatosis cerebri in children: A poor prognostic phenotype of diffuse gliomas with a distinct molecular profile"],"prefix":"10.1093","volume":"26","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0397-935X","authenticated-orcid":false,"given":"Gunther","family":"Nussbaumer","sequence":"first","affiliation":[{"name":"Division of Pediatric Hematology and Oncology, Department of Pediatrics and Adolescent Medicine, Medical University of Graz , Graz , Austria"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin","family":"Benesch","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology and Oncology, Department of Pediatrics and Adolescent Medicine, Medical University of Graz , Graz , Austria"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yura","family":"Grabovska","sequence":"additional","affiliation":[{"name":"Division of Molecular Pathology, Institute of Cancer Research , London , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alan","family":"Mackay","sequence":"additional","affiliation":[{"name":"Division of Molecular Pathology, Institute of Cancer Research , London , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Castel","sequence":"additional","affiliation":[{"name":"U981, Molecular Predictors and New Targets in Oncology, Team Genomics and Oncogenesis of Pediatric Brain Tumors, INSERM, Gustave Roussy, Universit\u00e9 Paris-Saclay , Villejuif , France"},{"name":"Department of Pediatric and Adolescent Oncology, Gustave Roussy, Universit\u00e9 Paris-Saclay , Villejuif , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacques","family":"Grill","sequence":"additional","affiliation":[{"name":"U981, Molecular Predictors and New Targets in Oncology, Team Genomics and Oncogenesis of Pediatric Brain Tumors, INSERM, Gustave Roussy, Universit\u00e9 Paris-Saclay , Villejuif , France"},{"name":"Department of Pediatric and Adolescent Oncology, Gustave Roussy, Universit\u00e9 Paris-Saclay , Villejuif , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marta M","family":"Alonso","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Cl\u00ednica Universidad de Navarra , Pamplona , Spain"},{"name":"Solid Tumor Program, Center for the Applied Medical Research , Pamplona, Navarra , Spain"},{"name":"Health Research Institute of Navarra (IdiSNA) , Pamplona, Navarra , Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manila","family":"Antonelli","sequence":"additional","affiliation":[{"name":"Department of Radiological, Oncological and Anatomo-Pathological Sciences, Sapienza University , Rome , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Simon","family":"Bailey","sequence":"additional","affiliation":[{"name":"Department of Paediatric Oncology, Sir James Spence Institute of Child Health , Royal Victoria Infirmary Queen Victoria Road, Newcastle upon Tyne , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7281-6306","authenticated-orcid":false,"given":"Joshua N","family":"Baugh","sequence":"additional","affiliation":[{"name":"Princess M\u00e1xima Center for Pediatric Oncology , Utrecht , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Veronica","family":"Biassoni","sequence":"additional","affiliation":[{"name":"Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale dei Tumori , Milano , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mirjam","family":"Blattner-Johnson","sequence":"additional","affiliation":[{"name":"Division of Pediatric Glioma Research, German Cancer Research Center (DKFZ) and German Cancer Consortium (DKTK) , Heidelberg , Germany"},{"name":"Hopp Children\u2019s Cancer Center (KiTZ) , Heidelberg , 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Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Esther","family":"Hulleman","sequence":"additional","affiliation":[{"name":"Princess M\u00e1xima Center for Pediatric Oncology , Utrecht , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisa","family":"Izquierdo","sequence":"additional","affiliation":[{"name":"Division of Molecular Pathology, Institute of Cancer Research , London , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sandra","family":"Jacobs","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology and Oncology, University Hospitals Leuven , Belgium"},{"name":"Pediatric Oncology, Department of Oncology , KU Leuven , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Karremann","sequence":"additional","affiliation":[{"name":"Department of Pediatric and Adolescent Medicine, University Medical Center Mannheim, Medical Faculty Mannheim, Heidelberg University , Mannheim , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonis","family":"Kattamis","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology-Oncology, First Department of Pediatrics, National and Kapodistrian University of Athens, \u201cAghia Sophia\u201d Children\u2019s Hospital , Athens , Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rejin","family":"Kebudi","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology-Oncology, Institute of Oncology, Istanbul University , Istanbul , Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8952-1819","authenticated-orcid":false,"given":"Rolf-Dieter","family":"Kortmann","sequence":"additional","affiliation":[{"name":"Department of Radiation Oncology, University of Leipzig , Leipzig , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert","family":"Kwiecien","sequence":"additional","affiliation":[{"name":"Institute of Biostatistics and Clinical Research, University of M\u00fcnster , M\u00fcnster , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maura","family":"Massimino","sequence":"additional","affiliation":[{"name":"Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale dei Tumori , Milano , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4408-2373","authenticated-orcid":false,"given":"Angela","family":"Mastronuzzi","sequence":"additional","affiliation":[{"name":"Department of Onco-Hematology, Gene and Cell Therapy, Bambino Ges\u00f9 Children\u2019s Hospital (IRCCS) , Rome , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Evelina","family":"Miele","sequence":"additional","affiliation":[{"name":"Department of Onco-Hematology, Gene 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