Targeting of the Hedgehog/GLI and mTOR pathways in advanced pancreatic cancer, a phase 1 trial of Vismodegib and Sirolimus combination.


Journal

Pancreatology : official journal of the International Association of Pancreatology (IAP) ... [et al.]
ISSN: 1424-3911
Titre abrégé: Pancreatology
Pays: Switzerland
ID NLM: 100966936

Informations de publication

Date de publication:
Sep 2020
Historique:
received: 17 04 2020
revised: 19 06 2020
accepted: 20 06 2020
pubmed: 12 8 2020
medline: 27 8 2021
entrez: 12 8 2020
Statut: ppublish

Résumé

Preclinical data indicated a functional and molecular interaction between Hedgehog (HH)/GLI and PI3K-AKT-mTOR pathways promoting pancreatic ductal adenocarcinoma (PDAC). A phase I study was conducted of Vismodegib and Sirolimus combination to evaluate maximum tolerated dose (MTD) and preliminary anti-tumor efficacy. Cohort I included advanced solid tumors patients following a traditional 3 + 3 design. Vismodegib was orally administered at 150 mg daily with Sirolimus starting at 3 mg daily, increasing to 6 mg daily at dose level 2. Cohort II included only metastatic PDAC patients. Anti-tumor efficacy was evaluated every two cycles and target assessment at pre-treatment and after a single cycle. Nine patient were enrolled in cohort I and 22 patients in cohort II. Twenty-eight patients were evaluated for dose-limiting toxicities (DLTs). One DLT was observed in each cohort, consisting of grade 2 mucositis and grade 3 thrombocytopenia. The MTD for Vismodegib and Sirolimus were 150 mg daily and 6 mg daily, respectively. The most common grade 3-4 toxicities were fatigue, thrombocytopenia, dehydration, and infections. A total of 6 patients had stable disease. No partial or complete responses were observed. Paired biopsy analysis before and after the first cycle in cohort II consistently demonstrated reduced GLI1 expression. Conversely, GLI and mTOR downstream targets were not significantly affected. The combination of Vismodegib and Sirolimus was well tolerated. Clinical benefit was limited to stable disease in a subgroup of patients. Targeting efficacy demonstrated consistent partial decreases in HH/GLI signaling with limited impact on mTOR signaling. These findings conflict with pre-clinical models and warrant further investigations.

Sections du résumé

BACKGROUND/OBJECTIVES OBJECTIVE
Preclinical data indicated a functional and molecular interaction between Hedgehog (HH)/GLI and PI3K-AKT-mTOR pathways promoting pancreatic ductal adenocarcinoma (PDAC). A phase I study was conducted of Vismodegib and Sirolimus combination to evaluate maximum tolerated dose (MTD) and preliminary anti-tumor efficacy.
METHODS METHODS
Cohort I included advanced solid tumors patients following a traditional 3 + 3 design. Vismodegib was orally administered at 150 mg daily with Sirolimus starting at 3 mg daily, increasing to 6 mg daily at dose level 2. Cohort II included only metastatic PDAC patients. Anti-tumor efficacy was evaluated every two cycles and target assessment at pre-treatment and after a single cycle.
RESULTS RESULTS
Nine patient were enrolled in cohort I and 22 patients in cohort II. Twenty-eight patients were evaluated for dose-limiting toxicities (DLTs). One DLT was observed in each cohort, consisting of grade 2 mucositis and grade 3 thrombocytopenia. The MTD for Vismodegib and Sirolimus were 150 mg daily and 6 mg daily, respectively. The most common grade 3-4 toxicities were fatigue, thrombocytopenia, dehydration, and infections. A total of 6 patients had stable disease. No partial or complete responses were observed. Paired biopsy analysis before and after the first cycle in cohort II consistently demonstrated reduced GLI1 expression. Conversely, GLI and mTOR downstream targets were not significantly affected.
CONCLUSIONS CONCLUSIONS
The combination of Vismodegib and Sirolimus was well tolerated. Clinical benefit was limited to stable disease in a subgroup of patients. Targeting efficacy demonstrated consistent partial decreases in HH/GLI signaling with limited impact on mTOR signaling. These findings conflict with pre-clinical models and warrant further investigations.

Identifiants

pubmed: 32778368
pii: S1424-3903(20)30213-1
doi: 10.1016/j.pan.2020.06.015
pii:
doi:

Substances chimiques

Anilides 0
Hedgehog Proteins 0
HhAntag691 0
Immunosuppressive Agents 0
Pyridines 0
RNA, Neoplasm 0
MTOR protein, human EC 2.7.1.1
TOR Serine-Threonine Kinases EC 2.7.11.1
Sirolimus W36ZG6FT64

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1115-1122

Informations de copyright

Copyright © 2020. Published by Elsevier B.V.

Déclaration de conflit d'intérêts

Declaration of competing interest The authors declared no conflicts with this submission.

Auteurs

Ryan M Carr (RM)

Schulze Center for Novel Therapeutics, Division of Oncology Research, Department of Oncology, Mayo Clinic, Rochester, MN, USA; Department of Medical Oncology, Department of Oncology, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA.

Narjust Duma (N)

Division of Hematology, Medical Oncology and Palliative Care, Department of Medicine, University of Wisconsin, Madison, WI, USA.

Angela L McCleary-Wheeler (AL)

Schulze Center for Novel Therapeutics, Division of Oncology Research, Department of Oncology, Mayo Clinic, Rochester, MN, USA.

Luciana L Almada (LL)

Schulze Center for Novel Therapeutics, Division of Oncology Research, Department of Oncology, Mayo Clinic, Rochester, MN, USA.

David L Marks (DL)

Schulze Center for Novel Therapeutics, Division of Oncology Research, Department of Oncology, Mayo Clinic, Rochester, MN, USA.

Rondell P Graham (RP)

Department of Laboratory Medicine Pathology, Mayo Clinic, Rochester, MN, USA.

Thomas C Smyrk (TC)

Department of Laboratory Medicine Pathology, Mayo Clinic, Rochester, MN, USA.

Val Lowe (V)

Department of Radiology, Mayo Clinic, Rochester, MN, USA.

Mitesh J Borad (MJ)

Division of Hematology-Medical Oncology, Mayo Clinic, Scottsdale, AZ, USA.

George Kim (G)

Division of Hematology-Oncology, The George Washington University, Washington, DC, USA.

Geoffrey B Johnson (GB)

Division of Nuclear Medicine, Mayo Clinic, Rochester, MN, USA.

Jacob B Allred (JB)

Department of Health Sciences Research, Mayo Clinic, Rochester, MN, USA.

Jun Yin (J)

Department of Health Sciences Research, Mayo Clinic, Rochester, MN, USA.

Vun-Sin Lim (VS)

Department of Medical Oncology, Department of Oncology, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA.

Tanios Bekaii-Saab (T)

Division of Hematology-Medical Oncology, Mayo Clinic, Scottsdale, AZ, USA.

Wen We Ma (WW)

Department of Medical Oncology, Department of Oncology, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA.

Charles Erlichman (C)

Department of Medical Oncology, Department of Oncology, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA.

Alex A Adjei (AA)

Department of Medical Oncology, Department of Oncology, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA. Electronic address: adjei.alex@mayo.edu.

Martin E Fernandez-Zapico (ME)

Schulze Center for Novel Therapeutics, Division of Oncology Research, Department of Oncology, Mayo Clinic, Rochester, MN, USA; Department of Medical Oncology, Department of Oncology, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA. Electronic address: fernanadezzapico.martin@mayo.edu.

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Classifications MeSH