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Record W4415479010 · doi:10.1016/j.ccell.2025.09.014

RAF-independent MEK mutations drive refractory histiocytic neoplasms but respond to ERK inhibition

2025· article· en· W4415479010 on OpenAlexaff
Eli L. Diamond, Jean‐François Emile, T Fujino, Julien Haroche, Maxim I. Maron, Alexander M. Lewis, Jahan Rahman, Anne S. Reiner, Dana Bossert, Marc K. Rosenblum, Mariko Yabe, Kseniya Petrova‐Drus, Jasmine H. Francis, Veronica Rotemberg, Raajit K. Rampal, Sarah Yoo, Anthony F. Daniyan, Sonia Mahajan, Vaios Hatzoglou, Robert J. Young, Gary A. Ulaner, Wiebke Rösler, Oshrat Hershkovitz‐Rokah, Ofer Shpilberg, Roei David Mazor, Luke Y. C. Chen, Michael Singer, M. Adriana Cuibus, Salima Benbarche, Pu Zhang, Nina Fox, Cynthia Aparecida de Castro, Steven Tittley, Matthew T. Witkowski, Fleur Cohen‐Aubart, Louis Terriou, Maher Hanoun, N. Schleinitz, Gabriela Alejandra Sosa, Timo Hautala, Laure Farnault De Lassus, Neal Rosen, Omar Abdel‐Wahab, Benjamin H. Durham

Bibliographic record

VenueCancer Cell · 2025
Typearticle
Languageen
FieldMedicine
TopicHistiocytic Disorders and Treatments
Canadian institutionsDalhousie University
FundersNational Cancer InstituteNational Institutes of HealthApplebaum FoundationCycle for SurvivalMarie-Josée and Henry R. Kravis Center for Molecular OncologyWest Family FoundationNational Cancer CenterEdward P. Evans FoundationAmerican Society of Hematology
KeywordsMEK inhibitorMAPK/ERK pathwayHistiocytic sarcomaMutationRefractory (planetary science)KinaseProtein kinase ANeoplasm

Abstract

fetched live from OpenAlex

Histiocytic neoplasms are clonal disorders of the monocyte/macrophage lineage defined by mutations activating mitogen-activated protein kinase (MAPK) signaling. Recently, the MEK1/2 inhibitor cobimetinib was FDA-approved for patients with adult histiocytoses. Here, aided by a prospective registry of patients with histiocytoses (NCT03329274), we identify that MEK1/2 mutations which constitutively activate MEK independently of RAF are associated with worse progression-free survival with MEK1/2 inhibition as compared to patients with other MEK1/2 mutational classes. The most common RAF-independent MEK1 mutation (MEK1 E102_I103del ) drove a lethal histiocytic-like neoplasm in mice, which was sensitive to the ERK1/2 inhibitor ulixertinib. We subsequently treated five MEK1 E102_I103del -mutant patients with ulixertinib on prospective protocols, four of whom were refractory to MEK inhibition. Four of five patients experienced objective responses to ulixertinib. These data reveal the impact of oncogenic MEK mutations in vivo , identify patients with likelihood of resistance to MEK inhibition, and nominate ERK inhibition to overcome resistance to MEK inhibition in histiocytoses. • RAF-independent (class III) MEK1 mutations are common in histiocytoses patients • Class III MEK1/2 mutations are associated with MEK inhibitor disease progression • Class III MEK1 E102_I103del mutation drives aggressive myeloid neoplasms in mice • Class III MEK1-mutant histiocytoses patients and mice respond to ERK inhibition Diamond et al. find RAF-independent (“class III”) mutations in MEK1/2 kinases are common in patients with systemic histiocytic neoplasms, drive disease in a conditional knock-in mouse model, and associate with progression on FDA-approved MEK inhibitors. Patients with these mutations who progress on MEK inhibitors respond to the ERK inhibitor ulixertinib.

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: Not applicable · Consensus signal: Not applicable
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.280
Threshold uncertainty score0.913

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)0.0010.000

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 head0.010
GPT teacher head0.288
Teacher spread0.279 · 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 designNot applicable
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

Citations3
Published2025
Admission routes1
Has abstractyes

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