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The type II RAF inhibitor tovorafenib in relapsed/refractory pediatric low-grade glioma: the phase 2 FIREFLY-1 trial

Title: The type II RAF inhibitor tovorafenib in relapsed/refractory pediatric low-grade glioma: the phase 2 FIREFLY-1 trial
Authors: Kilburn, LB; Khuong-Quang, DA; Hansford, JR; Landi, D; van der Lugt, J; Leary, SES; Driever, PH; Bailey, S; Perreault, S; McCowage, G; Waanders, AJ; Ziegler, DS; Witt, O; Baxter, PA; Kang, HJ; Hassall, TE; Han, JW; Hargrave, D; Franson, AT; Yalon Oren, M; Toledano, H; Larouche, V; Kline, C; Abdelbaki, MS; Jabado, N; Gottardo, NG; Gerber, NU; Whipple, NS; Segal, D; Chi, SN; Oren, L; Tan, EEK; Mueller, S; Cornelio, I; McLeod, L; Zhao, X; Walter, A; Da Costa, D; Manley, P; Blackman, SC; Packer, RJ; Nysom, K
Source: Nature Medicine (2023) (In press).
Publisher Information: Springer Science and Business Media LLC
Publication Year: 2023
Collection: University College London: UCL Discovery
Subject Terms: Drug development; Paediatric cancer; Phase II trials; Randomized controlled trials; Targeted therapies
Description: BRAF genomic alterations are the most common oncogenic drivers in pediatric low-grade glioma (pLGG). Arm 1 (n = 77) of the ongoing phase 2 FIREFLY-1 (PNOC026) trial investigated the efficacy of the oral, selective, central nervous system–penetrant, type II RAF inhibitor tovorafenib (420 mg m−2 once weekly; 600 mg maximum) in patients with BRAF-altered, relapsed/refractory pLGG. Arm 2 (n = 60) is an extension cohort, which provided treatment access for patients with RAF-altered pLGG after arm 1 closure. Based on independent review, according to Response Assessment in Neuro-Oncology High-Grade Glioma (RANO-HGG) criteria, the overall response rate (ORR) of 67% met the arm 1 prespecified primary endpoint; median duration of response (DOR) was 16.6 months; and median time to response (TTR) was 3.0 months (secondary endpoints). Other select arm 1 secondary endpoints included ORR, DOR and TTR as assessed by Response Assessment in Pediatric Neuro-Oncology Low-Grade Glioma (RAPNO) criteria and safety (assessed in all treated patients and the primary endpoint for arm 2, n = 137). The ORR according to RAPNO criteria (including minor responses) was 51%; median DOR was 13.8 months; and median TTR was 5.3 months. The most common treatment-related adverse events (TRAEs) were hair color changes (76%), elevated creatine phosphokinase (56%) and anemia (49%). Grade ≥3 TRAEs occurred in 42% of patients. Nine (7%) patients had TRAEs leading to discontinuation of tovorafenib. These data indicate that tovorafenib could be an effective therapy for BRAF-altered, relapsed/refractory pLGG. ClinicalTrials.gov registration: NCT04775485.
Document Type: article in journal/newspaper
File Description: text
Language: English
Relation: https://discovery.ucl.ac.uk/id/eprint/10182485/1/Hargrave_The%20type%20II%20RAF%20inhibitor%20tovorafenib%20in%20relapsed_VoR.pdf; https://discovery.ucl.ac.uk/id/eprint/10182485/
Availability: https://discovery.ucl.ac.uk/id/eprint/10182485/1/Hargrave_The%20type%20II%20RAF%20inhibitor%20tovorafenib%20in%20relapsed_VoR.pdf; https://discovery.ucl.ac.uk/id/eprint/10182485/
Rights: open
Accession Number: edsbas.B042311F
Database: BASE