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Record W4415963618 · doi:10.1038/s44276-025-00192-9

Somatic DICER1 pathogenic variants detected in a well-differentiated fetal lung adenocarcinoma diagnosed in a man with familial adenomatous polyposis

2025· article· en· W4415963618 on OpenAlexafffund
Afrida Ahmed, Paul S. Thorner, Anne‐Laure Chong, Naciba Benlimame, David Szlachtycz, George Chong, Sophie Camilleri‐Broët, William D. Foulkes

Bibliographic record

VenueBJC Reports · 2025
Typearticle
Languageen
FieldMedicine
TopicCongenital Diaphragmatic Hernia Studies
Canadian institutionsMcGill UniversityJewish General HospitalUniversity of TorontoMcGill University Health Centre
FundersCanadian Institutes of Health ResearchMerck CanadaPfizer
KeywordsFamilial adenomatous polyposisGermlineSomatic cellAdenomatous polyposis coliGermline mutationAdenocarcinomaLungEtiology

Abstract

fetched live from OpenAlex

Well-differentiated fetal lung adenocarcinoma (WDFLAC) is a rare pulmonary carcinoma characterized by its resemblance to fetal lung. It is thought to be caused by somatic pathogenic variants (PVs) in the genes CTNNB1 or APC, which lead to the nuclear accumulation of beta(β)-catenin and down-stream activation of key oncogenes. Recent studies, however, have shown that germline and/or somatic PVs in DICER1 are also implicated in the etiology of lung tumors with fetal-type morphologies. Here, we report the second case of WDFLAC in a male non-smoker with familial adenomatous polyposis (FAP), and the first case with confirmed inactivating PVs in both APC and DICER1. A review of the literature also showed that out of the 17 WDFLAC cases for which sequencing of DICER1, CTNNB1 and/or APC was performed, 13 had PVs in DICER1 and either CTNNB1 or APC, with one patient having all three genes mutated. Taken together, this suggests that the development of WDFLAC may be the summative effect of WNT and DICER1 dysfunction. It may thus be beneficial to test for DICER1 variants in diagnostically challenging cases and, if a germline DICER1 PV is found, additional screening for DICER1-associated tumors could be pursued.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.031
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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 head0.006
GPT teacher head0.235
Teacher spread0.229 · 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 teacher head, not a consensus.

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".

Quick stats

Citations0
Published2025
Admission routes2
Has abstractyes

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