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Record W3110760136 · doi:10.1093/neuonc/noaa222.225

ETMR-22. TITLE: DEFINING THE CLINICAL AND PROGNOSTIC LANDSCAPE OF EMBRYONAL TUMORS WITH MULTI-LAYERED ROSETTES (ETMRs), A RARE BRAIN TUMOR REGISTRY (RBTC) STUDY

2020· article· en· W3110760136 on OpenAlexaff
Sara Khan, Palma Solano‐Páez, Tannu Suwal, Salma Al‐Karmi, Mei Lu, Ben Ho, Maryam Fouladi, Sarah Leary, Jean M. Mulcahy Levy, Álvaro Lassaletta, Eloy Rivas, Alyssa Reddy, G. Yancey Gillespie, Nalin Gupta, Michal Yalon-Oren, Laura Amariglio, Hideo Nakamura, Kuo-Sheng Wu, Tai‐Tong Wong, Young‐Shin Ra, Milena La Spina, Luca Massimi, Anna Maria Buccoliero, Jordan R. Hansford, Richard G. Grundy, Dariusz Adamek, Jason Fangusaro, David Scharnhorst, Donna L. Johnston, Lucie Lafay‐Cousin, Sandra Camelo‐Piragua, Nabil Kabbara, Amar Gajjar, Mahjouba Boutarbouch, Maria João Gil‐da‐Costa, Derek Hanson, Paul Wood, Maysa Al‐Hussaini, Nisreen Amayiri, Yin Wang, Daniel Catchpoole, Jean Michaud, Anne Bendel, Benjamin Ellezam, Nicolas Gerber, Ashley Plant, Jeffery Rubens, Christopher Dunham, Christopher L. Moertel, Andrew W. Walter, David S. Ziegler, Andrew Dodgshun, Nicholas G. Gottardo, Ahmet Muzaffer Demir, Ramya Ramanujachar, Eric H. Raabe, Shago Mary, Peter B. Dirks, Michael D. Taylor, Hwang Eugene, Holly Lindsey, Tarık Tihan, Mette Jorgensen, Christine Dahl, Sharon Y. Y. Low, Amy Smith, Lili‐Naz Hazrati, Jesse Kresak, Gino R. Somers, Enrica E. K. Tan, Andres Morales, Vicente Santa‐María, Cynthia Hawkins, Ute Bartels, Derek Stephens, Sumihito Nobusawa, Christelle Dufour, Franck Bourdeaut, Nicolás André, Éric Bouffet, Annie Huang

Bibliographic record

VenueNeuro-Oncology · 2020
Typearticle
Languageen
FieldMedicine
TopicBrain Metastases and Treatment
Canadian institutionsUniversité de MontréalCentre Hospitalier Universitaire Sainte-JustineAlberta Children's HospitalChildren's Hospital of Eastern OntarioUniversity of TorontoSickKids FoundationUniversity of British ColumbiaHospital for Sick ChildrenOccupational Cancer Research Centre
Fundersnot available
KeywordsMedicineDiseaseInternal medicineCohortOncologyMultivariate analysisCerebrumRadiation therapyPediatricsPathologyGastroenterologyCentral nervous system

Abstract

fetched live from OpenAlex

Abstract ETMR, an aggressive disease characterised by C19MC alterations, were previously categorised as various histologic diagnoses. The clinical spectrum and impact of conventional multi-modal therapy on this new WHO diagnostic category remains poorly understood as a majority of ~200 cases reported to date lack molecular confirmation. We undertook comprehensive clinico-pathologic studies of a large molecularly confirmed cohort to improve disease recognition and treatment approaches. Amongst 623 CNS-PNETs patients enrolled in the RBTC registry, 159 primary ETMRs were confirmed based on a combination of FISH (125), methylation analysis (88), SNP and RNAseq (32) analyses; 91% had C19MC amplification/gains/fusions, 9% lacked C19MC alterations but had global methylation features of ETMR NOS. ETMRs arose in young patients (median age 26 months) predominantly as localized disease (M0-72%, M2-3 -18%) at multiple locations including cerebrum (60%) cerebellum (18%), midline structures (6%); notably 10% were brainstem primaries mimicking DIPG. Uni-and multivariate analyses of clinical and treatment details of curative regimens available for 110 patients identified metastatic disease (p=0.002), brainstem locations(p=0.005), extent of surgery, receipt of multi-modal therapy including high dose chemotherapy and radiation (P<0.001) as significant treatment prognosticators, while C19MC status, age and gender were non-significant risk factors. Analyses of events in all patients showed respective EFS at 3 and 12 months of 84%(95%CI:77–91) and 37%(95%CI:20–41) and 4yr OS of 27%(95%CI:18–37) indicating despite intensified therapies ETMR is a rapidly progressive and fatal disease. Our comprehensive data on the largest cohort of molecularly-confirmed ETMRs provides a critical framework to guide current clinical management and development of clinical trials.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.003
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0010.001
Open science0.0000.001
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0020.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.043
GPT teacher head0.340
Teacher spread0.297 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

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Citations0
Published2020
Admission routes1
Has abstractyes

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