https://m2.mtmt.hu/ hun 2026-10-24 01:33 JournalArticle 32327650 APPROVED true Department of Pediatric Oncology and Hematology, Charité - Universitätsmedizin Berlin, Freie Universität Berlin, Humboldt-Universität zu Berlin, Berlin, Germany Division of Neuropathology and Neurochemistry, Department of Neurology, Medical University of Vienna, Vienna, Austria Hopp Children's Cancer Center (KiTZ), Heidelberg, Germany Division of Pediatric Neurooncology, German Cancer Research Center (DKFZ), German Cancer Consortium (DKTK), Heidelberg, Germany Faculty of Biosciences, Heidelberg University, Heidelberg, Germany The Queen Silvia Children's Hospital, Sahlgrenska University Hospital, Gothenburg, Sweden Pediatric OncoGenomics Unit, Children's University Hospital Niño Jesús, Madrid, Spain Department of Pediatrics, KU Leuven and University Hospitals Leuven, Leuven, Belgium Second Faculty of Medicine, Charles University and University Hospital Motol, Prague, Czech Republic Department of Oncology, The Children's Memorial Health Institute, University of Warsaw, Warsaw, Poland Department of Pediatric and Adolescent Oncology, Gustave Roussy Cancer Center, Villejuif, France INSERM, Molecular Predictors and New Targets in Oncology, Paris-Saclay University, Villejuif, France Princess Máxima Center for Pediatric Oncology, Utrecht, Netherlands Department of Pediatrics and Adolescent Medicine, Comprehensive Center for Pediatrics, Medical University of Vienna, Vienna, Austria Developmental Biology and Cancer Research and Teaching Department, UCL Great Ormond Street Institute of Child Health, London, United Kingdom Department of Histopathology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, United Kingdom Department of Oncology, University Children's Hospital, Zurich, Switzerland Pediatric Unit, Fondazione IRCCS Istituto Nazionale Dei Tumori, Milan, Italy Pediatric Oncology Department, University Hospital São João, Porto, Portugal 2nd Department of Pediatrics, Semmelweis University, Budapest, Hungary Department of Neuro-Oncology, Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology, Moscow, Russian Federation Department of Pediatrics and Adolescent Medicine, Rigshospitalet, Copenhagen, Denmark Department of Pathology, Rigshospitalet, Copenhagen University Hospital, Copenhagen, Denmark Pediatric Oncology Department, Hospital Sant Joan de Déu, Barcelona, Spain Paediatric Haematology and Oncology Division, Hospital Universitari Vall d'Hebron, Barcelona, Spain Department of Radiation Oncology, Yonsei Cancer Center, Yonsei University College of Medicine, Seoul, South Korea Division of Pediatric and Adolescent Medicine, Oslo University Hospital, Oslo, Norway Department of Pathology, University Medical Centre Utrecht, Utrecht, Netherlands Service of Hematology/Oncology, Hospital JP Garrahan, Buenos Aires, Argentina Department of Pathology, NYU Langone Health and School of Medicine, New York, NY, United States Department of Neurooncology, Russian Scientific Center of Radiology, Moscow, Russian Federation Department of Neuroradiology, Burdenko Neurosurgical Institute, Moscow, Russian Federation Department of Pediatric Hematology and Oncology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany Institute of Neuropathology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany Research Institute Children's Cancer Center Hamburg, Hamburg, Germany Institute of Translational Research, University of Liverpool, Liverpool, United Kingdom Institute of Biostatistics and Clinical Research, University of Muenster, Muenster, Germany Institute of Biometry and Clinical Epidemiology, Charité University Medicine, Berlin Institute of Health, Berlin, Germany Department of Pediatric Hematology-Oncology, Center for Cancer and Immunology Research and Neuroscience Research, Children's National Medical Center, Washington, DC, United States Pediatric Glioma Research, German Cancer Research Center (DKFZ), German Cancer Consortium (DKTK), Heidelberg, Germany Department of Pediatric Oncology, Hematology and Immunology, Heidelberg University Hospital, Heidelberg, Germany Department of Neuropathology, Heidelberg University Hospital, Heidelberg, Germany Clinical Cooperation Unit Neuropathology, German Cancer Research Center (DKFZ), Heidelberg, Germany Institute of Neuropathology, University Medical Center Zurich, Zurich, Switzerland Department of Neuropathology, Burdenko Neurosurgical Institute, Moscow, Russian Federation Department of Pathomorphology, Children's Memorial Health Institute, Warsaw, Poland Department of Pathology, Amsterdam University Medical Center/VUmc, Amsterdam, Netherlands Department of Radiological, Oncological and Anatomopathological Sciences, Sapienza University of Rome, Rome, Italy IRCCS Neuromed, Pozzilli (IS), Italy Division of Pathology, The Hospital for Sick Children, Toronto, Canada Inst Neurophysiopathol, CHU Timone, Service d'Anatomie Pathologique et de Neuropathologie, Aix-Marseille Univ, APHM, CNRS, INP, Marseille, France Department of Pathology, Johns Hopkins University, Baltimore, MD, United States Neuropathology Unit, Division of Pathology, Fondazione Policlinico Universitario A. Gemelli, IRCCS, Università Cattolica Del Sacro Cuore, Rome, Italy Department of Neuropathology, Charité University Medicine, Berlin Institute of Health, Berlin, Germany German Cancer Consortium (DKTK), Partner Site Berlin, German Cancer Research Center (DKFZ), Heidelberg, Germany Department of Neuropathology, DGNN Brain Tumor Reference Center, University of Bonn, DZNE German Center for Neurodegenerative Diseases, Bonn, Germany Export Date: 30 May 2025; Cited By: 29; Correspondence Address: K. Von Hoff; Department of Pediatric Oncology and Hematology, Charité - Universitätsmedizin Berlin, Freie Universität Berlin, Humboldt-Universität zu Berlin, Berlin, Germany; email: katja.von-hoff@charite.de; CODEN: NEURJ 0 false 2026-10-21T19:49:55.298+0000 2025-04-17T19:46:29.876+0000 2021-10-09T19:40:28.988+0000 true 10067876 Ladányi Gusztáv /api/admin/10067876 Admin true 2026-08-04T08:40:33.479+0000 true false true 24 24 /api/publicationtype/24 PublicationType 1 true 24 JournalArticle true 10000359 Study Group true 24 24 /api/publicationtype/24 PublicationType 1 true 24 JournalArticle /api/subtype/10000359 Sokszerzős vagy csoportos szerzőségű szakcikk SubType 104 true 10000359 true 1 /api/category/1 Category true 1 von Hoff, Katja Therapeutic implications of improved molecular diagnostics for rare CNS embryonal tumor entities: results of an international, retrospective study true true /api/journal/10000906 REVIEWED true false 10000906 false 10000906 true 1522-8517 1523-5866 Journal FOREIGN 23 9 1597 1611 1597 15 2021 Background. Only few data are available on treatment-associated behavior of distinct rare CNS embryonal tumor entities previously treated as "CNS-primitive neuroectodermal tumors" (CNS-PNET). Respective data on specific entities, including CNS neuroblastoma, FOXR2 activated (CNS NB-FOXR2), and embryonal tumors with multilayered rosettes (ETMR) are needed for development of differentiated treatment strategies.Methods. Within this retrospective, international study, tumor samples of clinically well-annotated patients with the original diagnosis of CNS-PNET were analyzed using DNA methylation arrays (n = 307). Additional cases (n = 66) with DNA methylation pattern of CNS NB-FOXR2 were included irrespective of initial histological diagnosis. Pooled clinical data (n = 292) were descriptively analyzed.Results. DNA methylation profiling of "CNS-PNET" classified 58 (19%) cases as ETMR, 57 (19%) as high-grade glioma (HGG), 36 (12%) as CNS NB-FOXR2, and 89(29%) cases were classified into 18 other entities. Sixty-seven (22%) cases did not show DNA methylation patterns similar to established CNS tumor reference classes. Best treatment results were achieved for CNS NB-FOXR2 patients (5-year PFS: 63% 7%, OS: 85% +/- 5%, n = 63), with 35/42 progression-free survivors after upfront craniospinal irradiation (CSI) and chemotherapy. The worst outcome was seen for ETMR and HGG patients with 5-year PFS of 18% +/- 6% and 22% +/- 7%, and 5-year OS of 24% +/- 6% and 25% +/- 7%, respectively.Conclusion. The historically reported poor outcome of CNS-PNET patients becomes highly variable when tumors are molecularly classified based on DNA methylation profiling. Patients with CNS NB-FOXR2 responded well to current treatments and a standard-risk CSI-based regimen may be prospectively evaluated. 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class="authors"> <img title="Forrásközlemény" style="float: left" src="/frontend/resources/grid/publication-core-icon.png"> <img title="Idézőközlemény" style="float: left" src="/frontend/resources/grid/publication-citation-icon.png"> <div class="autype autype0"> <span class="author-name" >von Hoff Katja ✉ </span> ;&nbsp;&nbsp;&nbsp; <span class="author-name" mtid="10000251"><a href="/gui2/?type=authors&mode=browse&sel=10000251" target="_blank">Garami Miklos (<span class="authorship-author-name">Garami Miklós</span> <span class="authorAux-mtmt"> Gyermekgyógyászat</span>) </a> </span> <span class="author-affil"><span title="Semmelweis Egyetem">SE</span>/<span title="Általános Orvostudományi Kar">AOK</span>/<span title="Klinikum">K</span>/II. Sz. Gyermekgyógyászati Klinika</span> ;&nbsp;&nbsp;&nbsp; <span class="author-name" mtid="10012832"><a href="/gui2/?type=authors&mode=browse&sel=10012832" target="_blank">Hauser Peter (<span class="authorship-author-name">Hauser Péter</span> <span class="authorAux-mtmt"> Gyermekkori központi idegrendszeri daganatok</span>) </a> </span> <span class="author-affil"><span title="Semmelweis Egyetem">SE</span>/<span title="Általános Orvostudományi Kar">AOK</span>/<span title="Klinikum">K</span>/II. Sz. Gyermekgyógyászati Klinika</span> ;&nbsp;&nbsp;&nbsp; et al. (61) </div> <div><a target="_blank" href="/api/publication/32327650?labelLang=hun">Összes szerző</a></div> </div> <div class="title"><a href="/gui2/?mode=browse&params=publication;32327650" target="_blank">Therapeutic implications of improved molecular diagnostics for rare CNS embryonal tumor entities: results of an international, retrospective study</a></div> <div> <span class="journal-title">NEURO-ONCOLOGY</span> <span class="journal-issn">(<a target="_blank" href="https://portal.issn.org/resource/ISSN/1522-8517">1522-8517</a> <a target="_blank" href="https://portal.issn.org/resource/ISSN/1523-5866">1523-5866</a>)</span>: <span class="journal-volume">23</span> <span class="journal-issue">9</span> <span class="page"> pp 1597-1611 </span> <span class="year">(2021)</span> </div> <div class="pub-footer"> <span class="language" xmlns="http://www.w3.org/1999/html">Nyelv: Angol | </span> <span class="identifiers"> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="10.1093/neuonc/noab136" target="_blank" href="https://doi.org/10.1093/neuonc/noab136"> DOI </a> </span> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="000695839900022" target="_blank" href="https://www.webofscience.com/wos/woscc/full-record/000695839900022"> WoS </a> </span> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="85115953177" target="_blank" href="http://www.scopus.com/record/display.url?origin=inward&eid=2-s2.0-85115953177"> Scopus </a> </span> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="34077956" target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve&db=PubMed&list_uids=34077956&dopt=Abstract"> PubMed </a> </span> </span> <OnlyViewableByAuthor><div class="ratings"> <div class="journal-subject">Folyóirat szakterülete: Scopus - Cancer Research&nbsp;&nbsp;&nbsp;SJR indikátor:&nbsp;D1</div> <div class="journal-subject">Folyóirat szakterülete: Scopus - Neurology (clinical)&nbsp;&nbsp;&nbsp;SJR indikátor:&nbsp;D1</div> <div class="journal-subject">Folyóirat szakterülete: Scopus - Oncology&nbsp;&nbsp;&nbsp;SJR indikátor:&nbsp;D1</div> </div></OnlyViewableByAuthor> <div class="publication-citation" style="margin-left: 0.5cm;"> <span title="Nyilvános idézőközlemények összesen, említések nélkül" class="citingPub-count">Nyilvános idéző összesen: 43</span> | Független: 29 | Függő: 14 | Nem jelölt: 0 | WoS jelölt: 36 | Scopus jelölt:&nbsp;43 | WoS/Scopus jelölt:&nbsp;43 | DOI jelölt:&nbsp;43 (Nem nyilvános:&nbsp;1) </div> <div class="publication-citation"> <a target="_blank" href="/api/publication?cond=citations.related;eq;32327650&sort=publishedYear,desc&sort=title"> Idézett közlemények száma: 4 </a> </div> <div class="mtid"><span class="long-pub-mtid">Közlemény: 32327650</span> | <span class="status-data status-APPROVED"> Nyilvános </span> Forrás Idéző | <span class="type-subtype">Folyóiratcikk ( Sokszerzős vagy csoportos szerzőségű szakcikk ) </span> | <span class="pub-category">Tudományos</span> | <span class="publication-sourceOfData">WoS-XML</span> </div> <div class="lastModified">Utolsó módosítás: 2025.04.17. 21:46 Pécsi Éva (MTMT Közp 2,3, admin) </div> <pre class="comment" style="margin-top: 0; margin-bottom: 0;"><u>Megjegyzés</u>: Department of Pediatric Oncology and Hematology, Charité - Universitätsmedizin Berlin, Freie Universität Berlin, Humboldt-Universität zu Berlin, Berlin, Germany Division of Neuropathology and Neurochemistry, Department of Neurology, Medical University of Vienna, Vienna, Austria Hopp Children's Cancer Center (KiTZ), Heidelberg, Germany ...</pre> </div></div>