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We re-analyzed the series after a median of 119 months, adding retrospectively patients\u2019 molecular features.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Follow-up of all patients was updated. DNA copy number analysis and gene-fusion detection could be completed for 94\/160 patients, methylation classification for 68.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Progression-free survival (PFS) and overall survival (OS) at 5\/10 years were 66\/58%, and 80\/73%. Ten patients had late relapses (range 66\u2013126 mo), surviving after relapse no longer than those relapsing earlier (0\u20135 y). On multivariable analysis a better PFS was associated with grade II tumor and complete surgery at diagnosis and\/or at radiotherapy; female sex and complete resection showed a positive association with OS. Posterior fossa (PF) tumors scoring \u22650.80 on DNA methylation analysis were classified as PFA (n = 41) and PFB (n = 9). PFB patients had better PFS and OS. Eighteen\/32 supratentorial tumors were classified as RELA, and 3 as other molecular entities (anaplastic PXA, LGG MYB, HGNET). RELA had no prognostic impact. Patients with 1q gain or cyclin-dependent kinase inhibitor 2A (CDKN2A) loss had worse outcomes, included significantly more patients &amp;gt;3 years old (P = 0.050) and cases of dissemination at relapse (P = 0.007).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Previously described prognostic factors were confirmed at 10-year follow-up. Late relapses occurred in 6.2% of patients. Specific molecular features may affect outcome: PFB patients had a very good prognosis; 1q gain and CDKN2A loss were associated with dissemination. To draw reliable conclusions, modern ependymoma trials need to combine diagnostics with molecular risk stratification and long-term follow-up.<\/jats:p><\/jats:sec>","DOI":"10.1093\/neuonc\/noaa257","type":"journal-article","created":{"date-parts":[[2020,10,27]],"date-time":"2020-10-27T12:49:02Z","timestamp":1603802942000},"page":"848-857","source":"Crossref","is-referenced-by-count":37,"title":["Second series by the Italian Association of Pediatric Hematology and Oncology of children and adolescents with intracranial ependymoma: an integrated molecular and clinical characterization with a long-term follow-up"],"prefix":"10.1093","volume":"23","author":[{"given":"Maura","family":"Massimino","sequence":"first","affiliation":[{"name":"Departments of Pediatric, IRCCS Fondazione Istituto Nazionale dei Tumori, Milan, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesco","family":"Barretta","sequence":"additional","affiliation":[{"name":"Medical Statistics, Biometry and Bioinformatics, IRCCS Fondazione Istituto Nazionale dei Tumori, Milan, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Piergiorgio","family":"Modena","sequence":"additional","affiliation":[{"name":"Laboratory of Genetics, Sant\u2019Anna General Hospital, Como, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hendrik","family":"Witt","sequence":"additional","affiliation":[{"name":"Hopp-Children\u2019s Cancer Center Heidelberg, German Cancer Research Center, German Cancer Consortium , Heidelberg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Simone","family":"Minasi","sequence":"additional","affiliation":[{"name":"Departments of Neurology and Psychiatric, La Sapienza University, Rome, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefan M","family":"Pfister","sequence":"additional","affiliation":[{"name":"Hopp-Children\u2019s Cancer Center Heidelberg, German Cancer Research Center, German Cancer Consortium , Heidelberg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristian W","family":"Pajtler","sequence":"additional","affiliation":[{"name":"Hopp-Children\u2019s Cancer Center Heidelberg, German Cancer Research Center, German Cancer Consortium , Heidelberg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manila","family":"Antonelli","sequence":"additional","affiliation":[{"name":"Radiological, Oncological and Anatomo-Pathological Sciences, La Sapienza University, Rome, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lorenza","family":"Gandola","sequence":"additional","affiliation":[{"name":"Pediatric Radiotherapy, IRCCS Fondazione Istituto Nazionale dei Tumori, Milan, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"Luisa Garr\u00e8","sequence":"additional","affiliation":[{"name":"Neuroncology and Neurosurgery Unit, Giannina Gaslini Institute, Genova, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniele","family":"Bertin","sequence":"additional","affiliation":[{"name":"Pediatric Onco-Hematology, Units, Regina Margherita Children\u2019s Hospital, Torino, 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":"Pediatric Hematology and Oncology Department, Bambino Ges\u00f9 Pediatric Hospital, Rome, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maurizio","family":"Mascarin","sequence":"additional","affiliation":[{"name":"Pediatric Radiotherapy, Oncology Referral Center, Aviano, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lucia","family":"Quaglietta","sequence":"additional","affiliation":[{"name":"Departments of Pediatric Oncology, Santobono-Pausillipon Hospital, Naples, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisabetta","family":"Viscardi","sequence":"additional","affiliation":[{"name":"Pediatric Oncology, Padova University, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Iacopo","family":"Sardi","sequence":"additional","affiliation":[{"name":"Neuroncology, Units, Meyer Pediatric Hospital, Firenze, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio","family":"Ruggiero","sequence":"additional","affiliation":[{"name":"Pediatric Oncology, Units, A. 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