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Record W2953084859 · doi:10.1101/011304

Ovarian embryonal rhabdomyosarcoma is a rare manifestation of the DICER1 syndrome

2014· preprint· en· W2953084859 on OpenAlexafffund
Leanne de Kock, Harriet Druker, Evan Weber, Nancy Hamel, Jeffery Traubici, David Malkin, Jocelyne Arseneau, Dorothée Bouron‐Dal Soglio, John R. Priest, William D. Foulkes

Bibliographic record

VenuebioRxiv (Cold Spring Harbor Laboratory) · 2014
Typepreprint
Languageen
FieldMedicine
TopicCongenital Diaphragmatic Hernia Studies
Canadian institutionsUniversité de MontréalCentre Hospitalier Universitaire Sainte-JustineMcGill University Health CentreUniversity of TorontoSickKids FoundationHospital for Sick ChildrenMcGill UniversityJewish General Hospital
FundersCanadian Institutes of Health ResearchFaculty of Medicine, McGill UniversityMcGill UniversityAlex's Lemonade Stand Foundation for Childhood Cancer
KeywordsEmbryonal rhabdomyosarcomaRhabdomyosarcomaMedicinePathologySarcoma

Abstract

fetched live from OpenAlex

Abstract Embryonal rhabdomyosarcoma (ERMS), a malignant soft tissue sarcoma, is one of the most common paediatric cancers. Certain ERMS tumours are associated with the DICER1 syndrome, a distinctive tumour predisposition syndrome caused by germ-line mutations in the microRNA-maturation pathway gene, DICER1 . In addition to germ-line DICER1 mutations, highly characteristic somatic mutations have been identified in several DICER1-associated tumour types. These so-called “hotspot” mutations affect highly conserved amino acid residues central to the catalytic activity of the DICER1 ribonuclease IIIb domain. Primary ovarian ERMS (oERMS) is extremely rare. We present a case of a 6-year-old girl with an oERMS found to harbour two mutations in DICER1 . In addition to the oERMS, the girl also exhibited other DICER1 phenotypes, including cystic nephroma (CN) and multinodular goitre. Somatic investigations of the CN revealed the presence of a hotspot DICER1 mutation different from that in the oERMS. Of particular interest is the CN presented at the age of 12 years, which is much older than previously reported age range of susceptibility (birth to four years of age). This report documents both germ-line and highly characteristic somatic DICER1 mutations in a case of oERMS, adding to the expanding spectrum of rare childhood tumours in the DICER1 syndrome.

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)0.000
metaresearch head score (Gemma)0.001
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.000
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 head0.013
GPT teacher head0.224
Teacher spread0.212 · 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 designCase report
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".

Quick stats

Citations1
Published2014
Admission routes2
Has abstractyes

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