Pazopanib for treatment of typical solitary fibrous tumours: a multicentre, single-arm, phase 2 trial.


Journal

The Lancet. Oncology
ISSN: 1474-5488
Titre abrégé: Lancet Oncol
Pays: England
ID NLM: 100957246

Informations de publication

Date de publication:
03 2020
Historique:
received: 09 09 2019
revised: 22 11 2019
accepted: 10 12 2019
pubmed: 19 2 2020
medline: 7 7 2020
entrez: 19 2 2020
Statut: ppublish

Résumé

Solitary fibrous tumour is an ultra-rare sarcoma, which encompasses different clinicopathological subgroups. The dedifferentiated subgroup shows an aggressive course with resistance to pazopanib, whereas in the malignant subgroup, pazopanib shows higher activity than in previous studies with chemotherapy. We designed a trial to test pazopanib activity in two different cohorts of solitary fibrous tumour: the malignant-dedifferentiated cohort, which was previously published, and the typical cohort, which is presented here. In this single-arm, phase 2 trial, adult patients (aged ≥18 years) diagnosed with confirmed metastatic or unresectable typical solitary fibrous tumour of any location, who had progressed in the previous 6 months (by Choi criteria or Response Evaluation Criteria in Solid Tumors [RECIST]) and an Eastern Cooperative Oncology Group (ECOG) performance status of 0-2 were enrolled at 11 tertiary hospitals in Italy, France, and Spain. Patients received pazopanib 800 mg once daily, taken orally, until progression, unacceptable toxicity, withdrawal of consent, non-compliance, or a delay in pazopanib administration of longer than 3 weeks. The primary endpoint was proportion of patients achieving an overall response measured by Choi criteria in patients who received at least 1 month of treatment with at least one radiological assessment. All patients who received at least one dose of the study drug were included in the safety analyses. This study is registered in ClinicalTrials.gov, NCT02066285, and with the European Clinical Trials Database, EudraCT 2013-005456-15. From June 26, 2014, to Dec 13, 2018, of 40 patients who were assessed, 34 patients were enrolled and 31 patients were included in the response analysis. Median follow-up was 18 months (IQR 14-34), and 18 (58%) of 31 patients had a partial response, 12 (39%) had stable disease, and one (3%) showed progressive disease according to Choi criteria and central review. The proportion of overall response based on Choi criteria was 58% (95% CI 34-69). There were no deaths caused by toxicity, and the most frequent adverse events were diarrhoea (18 [53%] of 34 patients), fatigue (17 [50%]), and hypertension (17 [50%]). To our knowledge, this is the first prospective trial of pazopanib for advanced typical solitary fibrous tumour. The manageable toxicity and activity shown by pazopanib in this cohort suggest that this drug could be considered as first-line treatment for advanced typical solitary fibrous tumour. Spanish Group for Research on Sarcomas (GEIS), Italian Sarcoma Group (ISG), French Sarcoma Group (FSG), GlaxoSmithKline, and Novartis.

Sections du résumé

BACKGROUND
Solitary fibrous tumour is an ultra-rare sarcoma, which encompasses different clinicopathological subgroups. The dedifferentiated subgroup shows an aggressive course with resistance to pazopanib, whereas in the malignant subgroup, pazopanib shows higher activity than in previous studies with chemotherapy. We designed a trial to test pazopanib activity in two different cohorts of solitary fibrous tumour: the malignant-dedifferentiated cohort, which was previously published, and the typical cohort, which is presented here.
METHODS
In this single-arm, phase 2 trial, adult patients (aged ≥18 years) diagnosed with confirmed metastatic or unresectable typical solitary fibrous tumour of any location, who had progressed in the previous 6 months (by Choi criteria or Response Evaluation Criteria in Solid Tumors [RECIST]) and an Eastern Cooperative Oncology Group (ECOG) performance status of 0-2 were enrolled at 11 tertiary hospitals in Italy, France, and Spain. Patients received pazopanib 800 mg once daily, taken orally, until progression, unacceptable toxicity, withdrawal of consent, non-compliance, or a delay in pazopanib administration of longer than 3 weeks. The primary endpoint was proportion of patients achieving an overall response measured by Choi criteria in patients who received at least 1 month of treatment with at least one radiological assessment. All patients who received at least one dose of the study drug were included in the safety analyses. This study is registered in ClinicalTrials.gov, NCT02066285, and with the European Clinical Trials Database, EudraCT 2013-005456-15.
FINDINGS
From June 26, 2014, to Dec 13, 2018, of 40 patients who were assessed, 34 patients were enrolled and 31 patients were included in the response analysis. Median follow-up was 18 months (IQR 14-34), and 18 (58%) of 31 patients had a partial response, 12 (39%) had stable disease, and one (3%) showed progressive disease according to Choi criteria and central review. The proportion of overall response based on Choi criteria was 58% (95% CI 34-69). There were no deaths caused by toxicity, and the most frequent adverse events were diarrhoea (18 [53%] of 34 patients), fatigue (17 [50%]), and hypertension (17 [50%]).
INTERPRETATION
To our knowledge, this is the first prospective trial of pazopanib for advanced typical solitary fibrous tumour. The manageable toxicity and activity shown by pazopanib in this cohort suggest that this drug could be considered as first-line treatment for advanced typical solitary fibrous tumour.
FUNDING
Spanish Group for Research on Sarcomas (GEIS), Italian Sarcoma Group (ISG), French Sarcoma Group (FSG), GlaxoSmithKline, and Novartis.

Identifiants

pubmed: 32066540
pii: S1470-2045(19)30826-5
doi: 10.1016/S1470-2045(19)30826-5
pii:
doi:

Substances chimiques

Indazoles 0
Protein Kinase Inhibitors 0
Pyrimidines 0
Sulfonamides 0
pazopanib 7RN5DR86CK

Banques de données

ClinicalTrials.gov
['NCT02066285']

Types de publication

Clinical Trial, Phase II Journal Article Multicenter Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

456-466

Commentaires et corrections

Type : CommentIn
Type : CommentIn
Type : CommentIn

Informations de copyright

Copyright © 2020 Elsevier Ltd. All rights reserved.

Auteurs

Javier Martin-Broto (J)

Medical Oncology Department, University Hospital Virgen del Rocio, Seville, Spain; Institute of Biomedicine of Seville (University Hospital Virgen del Rocio; Spanish National Research Council; University of Seville), Seville, Spain. Electronic address: jmartin@mustbesevilla.org.

Josefina Cruz (J)

Medical Oncology Department, University Hospital of Canarias, Tenerife, Spain.

Nicolas Penel (N)

Department of Medical Oncology, Centre Oscar Lambret, Lille Medical School, Lille University, Lille, France.

Axel Le Cesne (A)

Gustave Roussy, Villejuif, France.

Nadia Hindi (N)

Medical Oncology Department, University Hospital Virgen del Rocio, Seville, Spain; Institute of Biomedicine of Seville (University Hospital Virgen del Rocio; Spanish National Research Council; University of Seville), Seville, Spain.

Pablo Luna (P)

Medical Oncology Department, University Hospital Son Espases, Mallorca, Spain.

David S Moura (DS)

Institute of Biomedicine of Seville (University Hospital Virgen del Rocio; Spanish National Research Council; University of Seville), Seville, Spain.

Daniel Bernabeu (D)

Radiology Department, University Hospital La Paz, Madrid, Spain.

Enrique de Alava (E)

Institute of Biomedicine of Seville (University Hospital Virgen del Rocio; Spanish National Research Council; University of Seville), Seville, Spain; Department of Normal and Pathological Cytology and Histology, School of Medicine, University of Seville, Seville, Spain; Centro de Investigación Biomédica en Red de Cáncer (CIBERONC), Seville, Spain.

Jose Antonio Lopez-Guerrero (JA)

Laboratory of Molecular Biology, Fundación Instituto Valenciano de Oncología, Valencia, Spain; Department of Basic Medical Sciences, School of Medicine, Catholic University of Valencia San Vicente Martir, Valencia, Spain.

Joaquin Dopazo (J)

Clinical Bioinformatics Area, Fundación Progreso y Salud, University Hospital Virgen del Rocio, Seville, Spain; Bioinformatics in Rare Diseases, Centro de Investigación Biomédica en Red de Enfermedades Raras, Fundación Progreso y Salud, University Hospital Virgen del Rocio, Seville, Spain; Spanish National Bioinformatics Institute, Fundación Progreso y Salud, Hospital Virgen del Rocío, Seville, Spain.

Maria Peña-Chilet (M)

Clinical Bioinformatics Area, Fundación Progreso y Salud, University Hospital Virgen del Rocio, Seville, Spain; Bioinformatics in Rare Diseases, Centro de Investigación Biomédica en Red de Enfermedades Raras, Fundación Progreso y Salud, University Hospital Virgen del Rocio, Seville, Spain.

Antonio Gutierrez (A)

Haematology Department, University Hospital Son Espases, Mallorca, Spain.

Paola Collini (P)

Soft Tissue and Bone Pathology, Histopathology and Pediatric Pathology Unit, Diagnostic Pathology and Laboratory Medicine Department, Fondazione Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Istituto Nazionale Tumori, Milan, Italy.

Marie Karanian (M)

Department of Biopathology Centre Leon Berard, Cancer Research Center of Lyon, Lyon, France.

Andres Redondo (A)

Department of Medical Oncology, University Hospital La Paz, Madrid, Spain.

Antonio Lopez-Pousa (A)

Medical Oncology Department, Sant Pau Hospital, Barcelona, Spain.

Giovanni Grignani (G)

Division of Medical Oncology, Candiolo Cancer Institute, Candiolo, Italy.

Juan Diaz-Martin (J)

Institute of Biomedicine of Seville (University Hospital Virgen del Rocio; Spanish National Research Council; University of Seville), Seville, Spain; Centro de Investigación Biomédica en Red de Cáncer (CIBERONC), Seville, Spain.

David Marcilla (D)

Department of Normal and Pathological Cytology and Histology, School of Medicine, University of Seville, Seville, Spain.

Antonio Fernandez-Serra (A)

Laboratory of Molecular Biology, Fundación Instituto Valenciano de Oncología, Valencia, Spain.

Cristina Gonzalez-Aguilera (C)

Institute of Biomedicine of Seville (University Hospital Virgen del Rocio; Spanish National Research Council; University of Seville), Seville, Spain.

Paolo G Casali (PG)

Cancer Medicine Department, Fondazione Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Istituto Nazionale Tumori, Milan, Italy.

Jean-Yves Blay (JY)

Centre Leon Berard, Université de Lyon, Lyon, France.

Silvia Stacchiotti (S)

Cancer Medicine Department, Fondazione Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Istituto Nazionale Tumori, Milan, Italy.

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Classifications MeSH