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Updated efficacy and safety of entrectinib in children with extracranial solid or primary central nervous system (CNS) tumors harboring <i>ROS1</i> fusions.

2025· article· en· W4410805450 on OpenAlexaff
Ami V. Desai, Huanmin Wang, Ellen M. Basu, Michela Casanova, Amy E. Armstrong, Daniel A. Morgenstern, Christine A. Pratilas, Alison K. Cardenas, Teresa Barata, Clare Devlin, Katherine E. Hutchinson, Felice Wu, Jade Wulff, Elizabeth Fox, Amar Gajjar

Bibliographic record

VenueJournal of Clinical Oncology · 2025
Typearticle
Languageen
FieldMedicine
TopicNeuroblastoma Research and Treatments
Canadian institutionsHospital for Sick Children
FundersF. Hoffmann-La Roche
KeywordsMedicineROS1Central nervous systemOncologyCancer researchCancerInternal medicineAdenocarcinoma

Abstract

fetched live from OpenAlex

10050 Background: Entrectinib is a TRK and ROS1 inhibitor that has shown rapid and durable responses in children with NTRK1/2/3 or ROS1 fusion-positive (fp) extracranial solid or primary CNS tumors in an integrated analysis of the STARTRK-NG (NCT02650401), TAPISTRY (NCT04589845) and STARTRK-2 (NCT02568267) trials. These data led to FDA and EMA approval of entrectinib in pediatric patients > 1 month with NTRK fp tumors. Here we present updated data on pediatric patients with ROS1 fp tumors based on the trials listed above to further describe the efficacy and safety of entrectinib in this population. Methods: Eligible pts were TRK/ROS1 inhibitor-naïve, < 18 years old, with locally advanced/metastatic extracranial solid or primary CNS tumors, with measurable or evaluable-only disease. All pts who received ≥1 daily dose of oral entrectinib are included in the safety-evaluable population. Pts who had a ROS1 fusion and were followed for ≥6 months are included in the ROS1 efficacy-evaluable population. Pts received entrectinib until disease progression, unacceptable toxicity, or consent withdrawal. Tumor responses were confirmed by blinded independent central review (BICR) per RECIST v1.1 or RANO criteria. Primary endpoint: confirmed objective response rate (ORR) per BICR. Key secondary endpoints: ORR in pts with baseline measurable disease per BICR; duration of confirmed response (DoR); time to confirmed response (TTR); clinical benefit rate (CBR); progression-free survival (PFS); overall survival (OS); safety. Results: At clinical cut-off (16 July 2024), of the 113 safety-evaluable pts, there were 26 pts in the ROS1 efficacy-evaluable cohort. ORR was 69.2% (95% CI 48.2, 85.7). Median TTR was 1.84 months. Median OS was not evaluable. Median duration of survival follow-up was 29.4 months (range 1–80). Efficacy outcomes are shown in the table. The most common related adverse events were weight gain (37.2%), anemia (36.3%), and AST increase (26.5%). Related fracture events occurred in 23% of pts. Conclusions: Entrectinib yielded rapid and durable responses in pediatric pts with ROS1 fp extracranial solid or primary CNS tumors. The safety profile of entrectinib was consistent with previous reports. Clinical trial information: NCT02650401 ; NCT04589845 ; NCT02568267 . Efficacy ROS1 (N=26) Confirmed ORR*, N, % [95% CI] 18, 69.2 [48.2- 85.7] Complete response 4, 15.4 [4.4- 34.9] Partial response 14, 53.8 [33.4- 73.4] Median confirmed DoR*, months (95% CI) NE (16.2- NE) Median TTR*, months (range) 1.84 (1.6- 4.0) CBR*, % (95% CI) 84.6 (65.1- 95.6) Median PFS*, months (95% CI) NE (21.8- NE) Median OS, months (95% CI) NE (NE- NE) *Per BICR; CI, confidence interval; NE, not evaluable.

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)0.002
metaresearch head score (Gemma)0.002
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Non-randomized trial · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.009

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0010.001
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0030.000

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 head0.034
GPT teacher head0.400
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 designNon-randomized trial
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".

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Citations0
Published2025
Admission routes1
Has abstractyes

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