REGISTRI: Regorafenib in first-line of KIT/PDGFRA wild type metastatic GIST: a collaborative Spanish (GEIS), Italian (ISG) and French Sarcoma Group (FSG) phase II trial.


Journal

Molecular cancer
ISSN: 1476-4598
Titre abrégé: Mol Cancer
Pays: England
ID NLM: 101147698

Informations de publication

Date de publication:
09 08 2023
Historique:
received: 08 06 2023
accepted: 26 07 2023
medline: 11 8 2023
pubmed: 10 8 2023
entrez: 9 8 2023
Statut: epublish

Résumé

Approximately 15% of adult GIST patients harbor tumors that are wild-type for KIT and PDGFRα genes (KP-wtGIST). These tumors usually have SDH deficiencies, exhibit a more indolent behavior and are resistant to imatinib. Underlying oncogenic mechanisms in KP-wtGIST include overexpression of HIF1α high IGFR signaling through the MAPK pathway or BRAF activating mutation, among others. As regorafenib inhibits these signaling pathways, it was hypothesized that it could be more active as upfront therapy in advanced KP-wtGIST. Adult patients with advanced KP-wtGIST after central confirmation by NGS, naïve of systemic treatment for advanced disease, were included in this international phase II trial. Eligible patients received regorafenib 160 mg per day for 21 days every 28 days. The primary endpoint was disease control rate (DCR), according to RECIST 1.1 at 12 weeks by central radiological assessment. From May 2016 to October 2020, 30 patients were identified as KP-wtGIST by Sanger sequencing and 16 were confirmed by central molecular screening with NGS. Finally, 15 were enrolled and received regorafenib. The study was prematurely closed due to the low accrual worsened by COVID outbreak. The DCR at 12 weeks was 86.7% by central assessment. A subset of 60% experienced some tumor shrinkage, with partial responses and stabilization observed in 13% and 87% respectively, by central assessment. SDH-deficient GIST showed better clinical outcome than other KP-wtGIST. Regorafenib activity in KP-wtGIST compares favorably with other tyrosine kinase inhibitors, especially in the SDH-deficient GIST subset and it should be taken into consideration as upfront therapy of advanced KP-wtGIST. ClinicalTrials.gov Identifier: NCT02638766.

Sections du résumé

BACKGROUND
Approximately 15% of adult GIST patients harbor tumors that are wild-type for KIT and PDGFRα genes (KP-wtGIST). These tumors usually have SDH deficiencies, exhibit a more indolent behavior and are resistant to imatinib. Underlying oncogenic mechanisms in KP-wtGIST include overexpression of HIF1α high IGFR signaling through the MAPK pathway or BRAF activating mutation, among others. As regorafenib inhibits these signaling pathways, it was hypothesized that it could be more active as upfront therapy in advanced KP-wtGIST.
METHODS
Adult patients with advanced KP-wtGIST after central confirmation by NGS, naïve of systemic treatment for advanced disease, were included in this international phase II trial. Eligible patients received regorafenib 160 mg per day for 21 days every 28 days. The primary endpoint was disease control rate (DCR), according to RECIST 1.1 at 12 weeks by central radiological assessment.
RESULTS
From May 2016 to October 2020, 30 patients were identified as KP-wtGIST by Sanger sequencing and 16 were confirmed by central molecular screening with NGS. Finally, 15 were enrolled and received regorafenib. The study was prematurely closed due to the low accrual worsened by COVID outbreak. The DCR at 12 weeks was 86.7% by central assessment. A subset of 60% experienced some tumor shrinkage, with partial responses and stabilization observed in 13% and 87% respectively, by central assessment. SDH-deficient GIST showed better clinical outcome than other KP-wtGIST.
CONCLUSIONS
Regorafenib activity in KP-wtGIST compares favorably with other tyrosine kinase inhibitors, especially in the SDH-deficient GIST subset and it should be taken into consideration as upfront therapy of advanced KP-wtGIST.
TRIAL REGISTRATION
ClinicalTrials.gov Identifier: NCT02638766.

Identifiants

pubmed: 37559050
doi: 10.1186/s12943-023-01832-9
pii: 10.1186/s12943-023-01832-9
pmc: PMC10413507
doi:

Substances chimiques

Antineoplastic Agents 0
Protein Kinase Inhibitors 0
Proto-Oncogene Proteins c-kit EC 2.7.10.1
regorafenib 24T2A1DOYB

Banques de données

ClinicalTrials.gov
['NCT02638766']

Types de publication

Clinical Trial, Phase II Letter Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

127

Informations de copyright

© 2023. BioMed Central Ltd., part of Springer Nature.

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Auteurs

Javier Martin-Broto (J)

Health Research Institute-Fundación Jiménez Díaz University Hospital, Universidad Autonoma de Madrid (IIS-FJD, UAM), 28040, Madrid, Spain. jmartin@atbsarc.org.
Medical Oncology Department, Fundación Jimenez Diaz University Hospital, Av. de los Reyes Católicos, 2, 28040, Madrid, Spain. jmartin@atbsarc.org.
General de Villalba University Hospital, 28400, Madrid, Spain. jmartin@atbsarc.org.

Claudia Valverde (C)

Medical Oncology department, Vall d'Hebron University Hospital, 08035, Barcelona, Spain.

Nadia Hindi (N)

Health Research Institute-Fundación Jiménez Díaz University Hospital, Universidad Autonoma de Madrid (IIS-FJD, UAM), 28040, Madrid, Spain.
Medical Oncology Department, Fundación Jimenez Diaz University Hospital, Av. de los Reyes Católicos, 2, 28040, Madrid, Spain.
General de Villalba University Hospital, 28400, Madrid, Spain.

Bruno Vincenzi (B)

Medical Oncology, University Campus Bio-Medico and Fondazione Policlinico Universitario Campus Bio-Medico, 00128, Rome, Italy.

Javier Martinez-Trufero (J)

Medical Oncology Department, University Hospital Miguel Servet, 50009, Zaragoza, Spain.

Giovanni Grignani (G)

Medical Oncology Unit, Città della Salute e della Scienza Hospital, 10126, Turin, Italy.

Antoine Italiano (A)

Medical Oncology department, Institute Bergonié, 33076, Bordeaux, France.

Javier Lavernia (J)

Medical Oncology department, Fundación Instituto Valenciano de Oncologia, 46009, Valencia, Spain.

Ana Vallejo (A)

Pathology department, Hospital Regional Universitario de Malaga, 29010, Malaga, Spain.

Paolo Dei Tos (PD)

Department of Medicine, School of Medicine, University of Padua, 35122, Padua, Italy.

Francois Le Loarer (F)

Pathology department, Institute Bergonié, 33076, Bordeaux, France.

Ricardo Gonzalez-Campora (R)

Pathology department, Hospital Quironsalud, 14004, Cordoba, Cordoba, Spain.

Rafael Ramos (R)

Pathology department, University Hospital Son Espases, 07120, Mallorca, Spain.

Diana Hernández-Jover (D)

Radiology department, Sant Pau University Hospital, 08025, Barcelona, Spain.

Antonio Gutierrez (A)

Hematology department, University Hospital Son Espases, 07120, Mallorca, Spain.

Cesar Serrano (C)

Medical Oncology department, Vall d'Hebron University Hospital, 08035, Barcelona, Spain.

Maria Monteagudo (M)

Hereditary Endocrine Cancer Group, Human Cancer Genetics Program, Spanish National Cancer Research Centre (CNIO), Madrid, Spain.

Rocio Letón (R)

Hereditary Endocrine Cancer Group, Human Cancer Genetics Program, Spanish National Cancer Research Centre (CNIO), Madrid, Spain.

Mercedes Robledo (M)

Hereditary Endocrine Cancer Group, Human Cancer Genetics Program, Spanish National Cancer Research Centre (CNIO), Madrid, Spain.
Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Institute of Health Carlos III (ISCIII), Madrid, Spain.

David S Moura (DS)

Health Research Institute-Fundación Jiménez Díaz University Hospital, Universidad Autonoma de Madrid (IIS-FJD, UAM), 28040, Madrid, Spain.

Marta Martin-Ruiz (M)

Health Research Institute-Fundación Jiménez Díaz University Hospital, Universidad Autonoma de Madrid (IIS-FJD, UAM), 28040, Madrid, Spain.

Jose A López-Guerrero (JA)

Molecular Biology department, Fundación Instituto Valenciano de Oncologia, 46009, Valencia, Spain.

Julia Cruz (J)

Pathology department, Fundación Instituto Valenciano de Oncologia, 46009, Valencia, Spain.

Antonio Fernandez-Serra (A)

Molecular Biology department, Fundación Instituto Valenciano de Oncologia, 46009, Valencia, Spain.

Jean-Yves Blay (JY)

Medicine Department, Centre Léon Bérard, 69008, Lyon, France.

Elena Fumagalli (E)

Medicine Department, Fondazione IRCCS - Istituto Nazionale dei Tumori, 20133, Milan, Italy.

Virginia Martinez-Marin (V)

Department of Medical Oncology, Hospital Universitario La Paz-IdiPAZ, P. Castellana, 261, 28046, Madrid, Spain.

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