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Record W4404149846 · doi:10.1093/bjd/ljae432

Exploration of the mutational landscape of cutaneous leiomyoma confirms <i>FH</i> as a driver gene and identifies targeting purine metabolism as a potential therapeutic strategy

2024· article· en· W4404149846 on OpenAlexfundno aff
Louise van der Weyden, Martin Del Castillo Velasco‐Herrera, Saamin Cheema, Kim Wong, Jacqueline Marcia Boccacino, Ian Vermes, Victoria Offord, Alastair Droop, David R A Jones, Elizabeth Anderson, Claire Hardy, Nicolas de Saint Aubain, Peter M. Ferguson, Carolin Mogler, Neil Rajan, Derek Frew, Paul W. Harms, Steven D. Billings, Désirée Schatton, Marc Segarra‐Mondejar, Mark J. Arends, Ingrid Ferreira, Thomas Brenn, Christian Frezza, David J. Adams

Bibliographic record

VenueBritish Journal of Dermatology · 2024
Typearticle
Languageen
FieldMedicine
TopicUrinary and Genital Oncology Studies
Canadian institutionsnot available
FundersMedical Research CouncilNational Institute for Health and Care ResearchMedical Research Council CanadaWellcome Trust
KeywordsPurineLeiomyomaGeneBiologyPurine analogueComputational biologyCancer researchBioinformaticsMedicineGeneticsPathologyBiochemistryEnzyme

Abstract

fetched live from OpenAlex

To comprehensively explore the mutational landscape of cutaneous leiomyoma (cLM) and identify candidate driver events, we performed a retrospective, multi-institutional, whole-exome sequencing and RNA sequencing study. We confirmed that a large proportion of patients with cLM have germline FH variants and additionally showed that somatic alteration of FH also drives cLM, with biallelic inactivation of FH being a frequent event. Treatment of Fh1-proficient and -deficient cell lines with the purine antagonist and chemotherapeutic agent, mercaptopurine, significantly decreased growth/colony formation; however, the addition of nucleosides was able to rescue only the Fh1-proficient cells, suggesting that purine metabolism is a targetable vulnerability for FH-deficient cLMs.

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)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.664
Threshold uncertainty score0.317

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
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 head0.013
GPT teacher head0.268
Teacher spread0.255 · 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.

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
Published2024
Admission routes1
Has abstractyes

Explore more

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