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A phase 2 randomized study of chemoimmunotherapy with or without eflornithine (DFMO) in relapsed/refractory neuroblastoma: A Children’s Oncology Group (COG) report.

2025· article· en· W4410802810 on OpenAlexaff
Margaret E. Macy, Arlene Naranjo, Fan F. Zhang, Michael R. Acord, Thomas F. Cash, Steven G. DuBois, Jennie Haunani Foster, Kristin R. Knight, Michael D. Hogarty, Rajen Mody, Daniel A. Morgenstern, Marguerite T. Parisi, Navin Pinto, C. Patrick Reynolds, Michelle E. Keyel, Barry L. Shulkin, David S. Ziegler, Rochelle Bagatell, Julie R. Park, Kelly C. Goldsmith

Bibliographic record

VenueJournal of Clinical Oncology · 2025
Typearticle
Languageen
FieldMedicine
TopicNeuroblastoma Research and Treatments
Canadian institutionsHospital for Sick Children
Fundersnot available
KeywordsMedicineChemoimmunotherapyCogEflornithineNeuroblastomaRefractory (planetary science)OncologyInternal medicineLomustineRandomized controlled trialChemotherapyVincristineCyclophosphamide

Abstract

fetched live from OpenAlex

10001 Background: Dinutuximab, irinotecan, temozolomide and GM-CSF (DIT) is widely used in first relapsed/ refractory high-risk neuroblastoma (r/r HRNB), however <50% of patients (pts) respond. ODC1 is a key enzyme important for NB cell survival. Difluoromethylornithine (DFMO) irreversibly inhibits ODC1, suppressing polyamine biosynthesis and driving anti-NB activity. DFMO also inhibits arginase and may enhance immunotherapy in r/r NB. COG ANBL1821 was a randomized phase 2 study for pts with r/r NB that evaluated response to DIT with or without DFMO (NCT03794349). Methods: Patients with first episode of r/r NB were randomized 1:1 to DIT (Arm A) or DIT with DFMO (6750 mg/m 2 divided TID; Arm B). They were stratified by measurable/evaluable disease, MYCN status, prior anti-GD2 therapy, and prior DFMO therapy. Cycles were 21 days. On Arm B, DFMO was initially given continuously; discontinuous DFMO dosing (days 1-7 and 15-21) was instituted due to ototoxicity in the initial cohort. Objective Response Rate [(ORR); sum of complete (CR), partial (PR), and minor (MR) responses per the 2017 International Neuroblastoma Response Criteria (INRC)] was determined based on central review of imaging. Toxicities were graded per CTCAE v5.0. Results: 91 eligible and evaluable pts (44 Arm A, 47 Arm B) were randomized May 2019-Jan 2024. 28/44 (63.6%) of Arm A pts and 32/47 (68.1%) of Arm B pts had relapsed or progressive disease (RPD); the remaining pts had refractory disease. The ORR was 61.4% (27/44) for Arm A and 57.4% (27/47) for Arm B (p=0.566). On Arm A, 22/44 (50%) achieved CR or PR compared to 23/27 (48.9%) on Arm B. Response rates for pts with RPD were similar to those with refractory disease in both arms. The 1-year progression-free survival (PFS) for Arm A was 70.0±8.0% and 56.8±8.2% for Arm B. Overall survival was 87.0±5.7% and 81.4±6.3% for Arms A and B, respectively. The most common toxicities reported on both arms were hematologic and gastrointestinal. Fewer pts on Arm B (n=7) had Grade 3+ pain than on Arm A (n=15) (p=0.0326). Hearing loss was a toxicity of interest; with continuous dosing, 55.6% (5/9) of Arm B pts developed hearing loss requiring DFMO dose hold. With discontinuous dosing, hearing loss was 15.8% (6/38) on Arm B compared to 6.3% (2/32) on Arm A (p=0.275). 14/16 (87.5%) NB+ marrows were GD2+ at enrollment; loss of GD2 was not detected on persisting NB cells in on therapy marrows analyzed. Conclusions: The addition of DFMO to DIT did not improve response rates in pts with first r/r HRNB, though the response rates in both arms confirmed DIT activity in this population. The ORR to DIT is higher than previously reported, likely due to use of the 2017 INRC that includes MR in calculation of ORR. Rates of CR+PR in this trial were similar to those previously reported. DFMO was associated with an increased incidence of hearing loss which was partially mitigated by discontinuous dosing. Clinical trial information: NCT03794349 .

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)0.001
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Randomized trial · Consensus signal: Randomized trial
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.006
Threshold uncertainty score0.021

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.001
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0020.001
Bibliometrics0.0000.000
Science and technology studies0.0000.001
Scholarly communication0.0010.001
Open science0.0010.000
Research integrity0.0020.003
Insufficient payload (model declined to judge)0.0060.001

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 head0.070
GPT teacher head0.492
Teacher spread0.422 · 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 designRandomized 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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Citations5
Published2025
Admission routes1
Has abstractyes

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