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Record W4406854676 · doi:10.1200/jco.24.00848

Elective Discontinuation of Larotrectinib in Pediatric Patients With TRK Fusion Sarcomas and Related Mesenchymal Tumors

2025· article· en· W4406854676 on OpenAlexaff
Leo Mascarenhas, Steven G. DuBois, Catherine M. Albert, Stefan Bielack, Daniel Orbach, Noah Federman, Birgit Geoerger, Ramamoorthy Nagasubramanian, Yizhuo Zhang, Julia Chisholm, Soledad Gallego, Hiroaki Goto, Daniel A. Morgenstern, Cormac Owens, Alberto S. Pappo, Sébastien Perreault, Johannes H. Schulte, Neerav Shukla, C. Michel Zwaan, Natascha Neu, Vadim Bernard-Gauthier, Esther De La Cuesta, Cornelis M. van Tilburg, Theodore W. Laetsch

Bibliographic record

VenueJournal of Clinical Oncology · 2025
Typearticle
Languageen
FieldMedicine
TopicSarcoma Diagnosis and Treatment
Canadian institutionsBayer (Canada)Centre Hospitalier Universitaire Sainte-JustineHospital for Sick ChildrenUniversity of Toronto
FundersNational Cancer InstituteLoxo OncologyBayer HealthCareJazz PharmaceuticalsEisaiGenentechY-mAbs TherapeuticsRoyal Marsden NHS Foundation TrustAmgenPfizerAstraZenecaEli Lilly and CompanyNational Institute for Health and Care ResearchAlex's Lemonade Stand Foundation for Childhood Cancer
KeywordsMedicineDiscontinuationTrk receptorSarcomaInternal medicineAdverse effectSurgeryOncologyPathologyReceptorNeurotrophin

Abstract

fetched live from OpenAlex

Larotrectinib is a highly selective tropomyosin receptor kinase (TRK) inhibitor with efficacy in children with TRK fusion tumors. We evaluated patient outcomes after elective discontinuation of larotrectinib in the absence of disease progression in a protocol-defined wait-and-see subset analysis of eligible patients where treatment resumption with larotrectinib was allowed if disease progressed. We also assessed the safety and efficacy of larotrectinib in all pediatric patients with sarcoma. This cohort included 91 patients (younger than 18 years) from two clinical trials: infantile fibrosarcoma (49), other soft tissue sarcomas or related mesenchymal tumors (41), and bone sarcoma (1). Treatment-related adverse events were of maximum grade 1 or 2 in 25% and 25% of patients, respectively. The overall response rate was 87% (95% CI, 78 to 93). In the wait-and-see analysis, 47 patients discontinued larotrectinib. Median time from discontinuation to disease progression was not reached. Sixteen patients had tumor progression during the wait-and-see period. All 16 patients resumed larotrectinib, and 15 (94%) achieved disease control, with 11 objective responses. Larotrectinib continues to demonstrate durable responses with favorable safety in children with TRK fusion sarcomas. Treatment discontinuation is feasible in select patients with objective response and clinical benefit noted in those who have disease progression after elective treatment discontinuation.

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)0.001
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: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.001
Threshold uncertainty score0.007

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
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 head0.030
GPT teacher head0.397
Teacher spread0.367 · 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

Citations13
Published2025
Admission routes1
Has abstractyes

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