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Record W3157748039 · doi:10.1210/jendso/bvab048.1363

Infigratinib in Children With Achondroplasia (ACH): Design of PROPEL2 - A Phase 2, Open-Label, Dose-Escalation and Dose-Expansion Study

2021· article· en· W3157748039 on OpenAlexaff
Ravi Savarirayan, Pekka Kannus, Carl Dambkowski, Daniela Rogoff, Melita Irving

Bibliographic record

VenueJournal of the Endocrine Society · 2021
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicConnective tissue disorders research
Canadian institutionsHospital for Sick Children
Fundersnot available
KeywordsMedicineEndochondral ossificationInternal medicineAdverse effectAchondroplasiaOncologyEndocrinologyPharmacologySurgeryAnatomyCartilage

Abstract

fetched live from OpenAlex

Abstract Background: ACH, the most common non-lethal form of skeletal dysplasia, is characterized by defective endochondral ossification resulting from gain-of-function mutations in the fibroblast growth factor receptor 3 (FGFR3) gene, a negative regulator of endochondral bone formation. Current treatment options are non-targeted, ineffective, or painful interventions aimed at preventing or treating complications. Infigratinib is an orally bioavailable and selective FGFR1-3 tyrosine kinase inhibitor in development for FGFR-related conditions. In vitro data with infigratinib showed inhibition of FGFR1-3 activity with reversal of established growth arrest in chondrocytes. In vivo studies revealed dose-dependent improvements in foramen magnum and long bone length in Fgfr3Y367C/+ mice following treatment with infigratinib. Methods: PROPEL2 is a prospective, phase 2, open-label study of infigratinib in children with ACH. Children 3-11 years of age with ACH who have completed at least 6 months of observation in the observational PROPEL study are eligible to participate. PROPEL2 consists of dose escalation with an extended treatment phase, designed as dose finding, followed by a dose-expansion phase to confirm the selected dose and to provide evidence of efficacy. The primary endpoints of the dose-escalation/extended treatment phase are treatment-emergent adverse events and change from baseline in annualized growth velocity. Subjects (n=40) will be enrolled in ascending dose cohorts of approximately 10 subjects/cohort (4 cohorts planned) and treated for 6 months at their assigned dose, continuing for an additional 12 months with dose modifications as required. Up to 20 new subjects will be enrolled in the dose-expansion phase and receive infigratinib (mini-tablets, administered orally once daily) for 12 months. Secondary objectives include: safety/tolerability of infigratinib; changes from baseline in anthropometric parameters, including body proportions; and the pharmacokinetic/pharmacodynamic profile of infigratinib. An exploratory objective is evaluation of changes in ACH disease burden. Current Status: PROPEL2 is currently enrolling - the first subject was entered in July 2020. The planned total enrollment is 60 children with ACH (n=40 in the dose-escalation/extended treatment phase; n=20 in the dose-expansion phase). Following completion of PROPEL2, subjects have the opportunity to enroll in an open-label long-term extension study to assess the safety and efficacy of long-term administration of infigratinib in children with ACH.

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)0.004
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 score0.004
Threshold uncertainty score0.020

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0040.002
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0030.001
Bibliometrics0.0010.001
Science and technology studies0.0000.001
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0020.003
Insufficient payload (model declined to judge)0.0040.001

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 head0.035
GPT teacher head0.354
Teacher spread0.319 · 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".

Quick stats

Citations0
Published2021
Admission routes1
Has abstractyes

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