{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T12:10:26Z","timestamp":1753877426213,"version":"3.41.2"},"reference-count":24,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2024,6,12]],"date-time":"2024-06-12T00:00:00Z","timestamp":1718150400000},"content-version":"vor","delay-in-days":163,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000054","name":"National Cancer Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000054","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,1,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>Choroid plexus tumors (CPTs) are rare, potentially aggressive CNS tumors with defined histologic criteria for grading. In recent years, several patients within our practice have demonstrated discordance between the histologic diagnosis and clinical behavior. DNA methylation profiling has emerged as a potential diagnostic adjunct for aiding the clinical approach.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>We reviewed the clinical and pathologic data of all CPTs diagnosed at Boston Children\u2019s Hospital from 1995 to 2023. All cases with available material (38\/48) underwent DNA methylation profiling at NIH\/NCI, and the classifier results were correlated with the WHO histologic grade and patient outcomes. Survival information was analyzed using Kaplan\u2013Meier curves.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>There was good correlation (11\/12, 92%) between methylation class and WHO histologic grade for choroid plexus carcinomas (CPC); one histologic CPC grouped with choroid plexus papilloma (CPP) group pediatric (P). Five CPPs grouped with methylation class CPC (5\/17, 29%). In the group of atypical CPPs (n\u2005=\u20059), there were two that grouped with methylation class CPC. Survival analysis showed utility of methylation classes in the prediction of biologic behavior.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>Results indicated that methylation profiling may serve as a valuable tool in the clinical decision-making process for patients with CPTs, providing additional prognostic information compared to WHO histologic grade alone. The value of methylation array analysis is particularly important given the lack of consensus on treatment regimens for CPTs.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/noajnl\/vdae097","type":"journal-article","created":{"date-parts":[[2024,6,11]],"date-time":"2024-06-11T22:01:35Z","timestamp":1718143295000},"source":"Crossref","is-referenced-by-count":0,"title":["Clinical utility of DNA methylation profiling for choroid plexus tumors"],"prefix":"10.1093","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0713-6384","authenticated-orcid":false,"given":"Kee Kiat","family":"Yeo","sequence":"first","affiliation":[{"name":"Department of Pediatric Oncology, Dana-Farber\/Boston Children\u2019s Cancer and Blood Disorder Center , Boston, Massachusetts ,\u00a0 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cassie B","family":"Macrae","sequence":"additional","affiliation":[{"name":"Department of Pathology, Boston Children\u2019s Hospital , Boston, Massachusetts ,\u00a0 USA"},{"name":"Department of Forensic Pathology, University of New Mexico , Albuquerque, New Mexico , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bradley","family":"Gampel","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology, Dana-Farber\/Boston Children\u2019s Cancer and Blood Disorder Center , Boston, Massachusetts ,\u00a0 USA"},{"name":"Department of Pediatrics, Sylvester Comprehensive Cancer Center , Miami, Florida , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jared T","family":"Ahrendsen","sequence":"additional","affiliation":[{"name":"Department of Pathology, Northwestern University Feinberg School of Medicine , Chicago, Illinois , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hart","family":"Lidov","sequence":"additional","affiliation":[{"name":"Department of Pathology, Boston Children\u2019s Hospital , Boston, Massachusetts ,\u00a0 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Karen D","family":"Wright","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology, Dana-Farber\/Boston Children\u2019s Cancer and Blood Disorder Center , Boston, Massachusetts ,\u00a0 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Susan","family":"Chi","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology, Dana-Farber\/Boston Children\u2019s Cancer and Blood Disorder Center , Boston, Massachusetts ,\u00a0 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Katie","family":"Fehnel","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Boston Children\u2019s Hospital , Boston, Massachusetts , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lissa","family":"Baird","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Boston Children\u2019s Hospital , Boston, Massachusetts , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jessica","family":"Clymer","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology, Dana-Farber\/Boston Children\u2019s Cancer and Blood Disorder Center , Boston, Massachusetts ,\u00a0 USA"},{"name":"Department of Pediatrics, New York University Lagone Medical Center , New York, New York , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5119-7550","authenticated-orcid":false,"given":"Kenneth","family":"Aldape","sequence":"additional","affiliation":[{"name":"Center for Cancer Research, National Cancer Institute , Bethesda, Maryland , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9246-4184","authenticated-orcid":false,"given":"Sanda","family":"Alexandrescu","sequence":"additional","affiliation":[{"name":"Department of Pathology, Boston Children\u2019s Hospital , Boston, Massachusetts ,\u00a0 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2024,6,12]]},"reference":[{"issue":"Suppl 5","key":"2024070313363497900_CIT0001","doi-asserted-by":"crossref","first-page":"v1","DOI":"10.1093\/neuonc\/noac202","article-title":"CBTRUS statistical report: primary brain and other central nervous system tumors diagnosed in the United States in 2015\u20132019","volume":"24","author":"Ostrom","year":"2022","journal-title":"Neuro Oncol"},{"key":"2024070313363497900_CIT0002","volume-title":"World Health Organization Classification of Tumours of the Central Nervous System","author":"WHO Classification of Tumours Editorial Board","year":"2021","edition":"5th ed"},{"issue":"5","key":"2024070313363497900_CIT0003","doi-asserted-by":"crossref","first-page":"398","DOI":"10.3171\/2012.8.PEDS12132","article-title":"Treatment of choroid plexus tumors: a 20-year single institutional experience","volume":"10","author":"Bettegowda","year":"2012","journal-title":"J Neurosurg Pediatr"},{"issue":"4","key":"2024070313363497900_CIT0004","doi-asserted-by":"crossref","first-page":"745","DOI":"10.1007\/s00701-019-03832-5","article-title":"Management of choroid plexus tumors\u2014an institutional experience","volume":"161","author":"Hosmann","year":"2019","journal-title":"Acta Neurochir (Wien)."},{"issue":"3","key":"2024070313363497900_CIT0005","doi-asserted-by":"crossref","first-page":"599","DOI":"10.1007\/s11060-021-03942-0","article-title":"Final results of the Choroid Plexus Tumor study CPT-SIOP-2000","volume":"156","author":"Wolff","year":"2022","journal-title":"J Neurooncol."},{"issue":"5","key":"2024070313363497900_CIT0006","doi-asserted-by":"crossref","first-page":"784","DOI":"10.1002\/pbc.25436","article-title":"Choroid plexus carcinoma in children: the Head Start experience","volume":"62","author":"Zaky","year":"2015","journal-title":"Pediatr Blood Cancer."},{"issue":"1","key":"2024070313363497900_CIT0007","first-page":"vdaa139","article-title":"Role of available adjuvant therapies following surgical resection of atypical choroid plexus","volume":"2","author":"Tavallaii","year":"2020","journal-title":"Neurooncol. Adv."},{"issue":"3","key":"2024070313363497900_CIT0008","doi-asserted-by":"crossref","first-page":"383","DOI":"10.1007\/s11060-009-9936-y","article-title":"Atypical choroid plexus papilloma: clinical experience in the CPT-SIOP-2000 study","volume":"95","author":"Wrede","year":"2009","journal-title":"J Neurooncol."},{"issue":"1","key":"2024070313363497900_CIT0009","doi-asserted-by":"crossref","first-page":"63","DOI":"10.1007\/s11060-021-03843-2","article-title":"Paediatric atypical choroid plexus papilloma: is adjuvant therapy necessary","volume":"155","author":"Browne-Farmer","year":"2021","journal-title":"J Neurooncol."},{"issue":"7697","key":"2024070313363497900_CIT0010","doi-asserted-by":"crossref","first-page":"469","DOI":"10.1038\/nature26000","article-title":"DNA methylation-based classification of central nervous system tumours","volume":"555","author":"Capper","year":"2018","journal-title":"Nature."},{"issue":"6","key":"2024070313363497900_CIT0011","doi-asserted-by":"crossref","first-page":"790","DOI":"10.1093\/neuonc\/nov322","article-title":"Methylation profiling of choroid plexus tumors reveals 3 clinically distinct subgroups","volume":"18","author":"Thomas","year":"2016","journal-title":"Neuro Oncol"},{"issue":"1","key":"2024070313363497900_CIT0012","doi-asserted-by":"crossref","first-page":"117","DOI":"10.1186\/s13148-019-0708-z","article-title":"DNA methylation signature is prognostic of choroid plexus tumor aggressiveness","volume":"11","author":"Pienkowska","year":"2019","journal-title":"Clin Epigenetics"},{"issue":"1","key":"2024070313363497900_CIT0013","first-page":"vdaa168","article-title":"Outcome and molecular analysis of young children with choroid plexus carcinoma treated with non-myeloablative therapy: results from the SJYC07 trial","volume":"3","author":"Liu","year":"2021","journal-title":"Neurooncol. Adv."},{"issue":"4","key":"2024070313363497900_CIT0014","doi-asserted-by":"crossref","first-page":"571","DOI":"10.1093\/neuonc\/noab227","article-title":"Impact of the methylation classifier and ancillary methods on CNS tumor diagnostics","volume":"24","author":"Wu","year":"2022","journal-title":"Neuro Oncol"},{"key":"2024070313363497900_CIT0015","first-page":"Article 28","article-title":"RAWGraphs: A Visualisation Platform to Create Open Outputs","author":"Mauri","year":"2017"},{"issue":"1","key":"2024070313363497900_CIT0016","doi-asserted-by":"crossref","first-page":"184","DOI":"10.1158\/1078-0432.CCR-14-1324","article-title":"Molecular characterization of choroid plexus tumors reveals novel clinically relevant subgroups","volume":"21","author":"Merino","year":"2015","journal-title":"Clin Cancer Res."},{"issue":"1","key":"2024070313363497900_CIT0017","doi-asserted-by":"crossref","first-page":"151","DOI":"10.1007\/s11060-017-2561-2","article-title":"A French retrospective study on clinical outcome in 102 choroid plexus tumors in children","volume":"135","author":"Siegfried","year":"2017","journal-title":"J Neurooncol."},{"issue":"6","key":"2024070313363497900_CIT0018","doi-asserted-by":"crossref","first-page":"615","DOI":"10.3171\/2010.3.PEDS09354","article-title":"Use of ifosfamide, carboplatin, and etoposide chemotherapy in choroid plexus carcinoma","volume":"5","author":"Lafay-Cousin","year":"2010","journal-title":"J Neurosurg Pediatr"},{"issue":"2","key":"2024070313363497900_CIT0019","doi-asserted-by":"crossref","first-page":"259","DOI":"10.1007\/s00381-010-1269-9","article-title":"Choroid plexus tumors in children less than 36 months: the Canadian Pediatric Brain Tumor Consortium (CPBTC) experience","volume":"27","author":"Lafay-Cousin","year":"2011","journal-title":"Childs Nerv Syst."},{"issue":"3","key":"2024070313363497900_CIT0020","doi-asserted-by":"crossref","first-page":"470","DOI":"10.1097\/00006123-199803000-00006","article-title":"Choroid plexus carcinomas in childhood: clinical features and prognostic factors","volume":"42","author":"Berger","year":"1998","journal-title":"Neurosurgery."},{"issue":"4","key":"2024070313363497900_CIT0021","doi-asserted-by":"crossref","first-page":"e844","DOI":"10.1097\/MPH.0000000000002428","article-title":"Prognostic implication of DNA methylation signature in atypical choroid plexus papilloma with intracranial dissemination","volume":"44","author":"Yanagi","year":"2022","journal-title":"J Pediatr Hematol Oncol."},{"issue":"3","key":"2024070313363497900_CIT0022","doi-asserted-by":"crossref","first-page":"127","DOI":"10.1159\/000028847","article-title":"Malignant evolution of choroid plexus papilloma","volume":"31","author":"Chow","year":"1999","journal-title":"Pediatr Neurosurg."},{"issue":"1","key":"2024070313363497900_CIT0023","first-page":"1","article-title":"[Carcinomas and malignant papillomas of choroid plexus (author\u2019s transl)]","volume":"22","author":"Gullotta","year":"1979","journal-title":"Neurochirurgia (Stuttg)"},{"issue":"4","key":"2024070313363497900_CIT0024","doi-asserted-by":"crossref","first-page":"321","DOI":"10.1007\/BF00697007","article-title":"Ultrastructure of disseminated choroid plexus papilloma","volume":"54","author":"Masuzawa","year":"1981","journal-title":"Acta Neuropathol."}],"container-title":["Neuro-Oncology Advances"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/noa\/advance-article-pdf\/doi\/10.1093\/noajnl\/vdae097\/58208984\/vdae097.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/noa\/article-pdf\/6\/1\/vdae097\/58412326\/vdae097.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/noa\/article-pdf\/6\/1\/vdae097\/58412326\/vdae097.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,7,3]],"date-time":"2024-07-03T13:37:11Z","timestamp":1720013831000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/noa\/article\/doi\/10.1093\/noajnl\/vdae097\/7691768"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,1,1]]},"references-count":24,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2024,1,1]]}},"URL":"https:\/\/doi.org\/10.1093\/noajnl\/vdae097","relation":{},"ISSN":["2632-2498"],"issn-type":[{"type":"electronic","value":"2632-2498"}],"subject":[],"published-other":{"date-parts":[[2024,1]]},"published":{"date-parts":[[2024,1,1]]},"article-number":"vdae097"}}