Identifying and targeting cancer stem cells in leiomyosarcoma: prognostic impact and role to overcome secondary resistance to PI3K/mTOR inhibition.


Journal

Journal of hematology & oncology
ISSN: 1756-8722
Titre abrégé: J Hematol Oncol
Pays: England
ID NLM: 101468937

Informations de publication

Date de publication:
25 01 2019
Historique:
received: 15 09 2018
accepted: 27 12 2018
entrez: 27 1 2019
pubmed: 27 1 2019
medline: 11 3 2020
Statut: epublish

Résumé

Leiomyosarcoma (LMS) is one of the most frequent soft tissue sarcoma subtypes and is characterized by a consistent deregulation of the PI3K/mTOR pathway. Cancer stem cells (CSCs) have been poorly studied in soft tissue sarcomas. In this study, we aimed to evaluate the association between CSCs, the outcome of LMS patients, and the resistance to PI3K/mTOR pathway inhibition. We investigated the relationships between aldehyde dehydrogenase 1 (ALDH1) expression, a cancer stem cell marker, and the outcome of LMS patients in two independent cohorts. We assessed the impact of CSCs in resistance to PI3K/mTOR pathway inhibition using LMS cell lines, a xenograft mouse model, and human tumor samples. We found that enhanced ALDH1 activity is a hallmark of LMS stem cells and is an independent prognostic factor. We also identified that secondary resistance to PI3K/mTOR pathway inhibition was associated with the expansion of LMS CSCs. Interestingly, we found that EZH2 inhibition, a catalytic component of polycomb repressive complex which plays a critical role in stem cell maintenance, restored sensitivity to PI3K/mTOR pathway inhibition. Importantly, we confirmed the clinical relevance of our findings by analyzing tumor samples from patients who showed secondary resistance after treatment with a PI3Kα inhibitor. Altogether, our findings suggest that CSCs have a strong impact on the outcome of patients with LMS and that combining PI3K/mTOR and EZH2 inhibitors may represent a promising strategy in this setting.

Sections du résumé

BACKGROUND
Leiomyosarcoma (LMS) is one of the most frequent soft tissue sarcoma subtypes and is characterized by a consistent deregulation of the PI3K/mTOR pathway. Cancer stem cells (CSCs) have been poorly studied in soft tissue sarcomas. In this study, we aimed to evaluate the association between CSCs, the outcome of LMS patients, and the resistance to PI3K/mTOR pathway inhibition.
METHODS
We investigated the relationships between aldehyde dehydrogenase 1 (ALDH1) expression, a cancer stem cell marker, and the outcome of LMS patients in two independent cohorts. We assessed the impact of CSCs in resistance to PI3K/mTOR pathway inhibition using LMS cell lines, a xenograft mouse model, and human tumor samples.
RESULTS
We found that enhanced ALDH1 activity is a hallmark of LMS stem cells and is an independent prognostic factor. We also identified that secondary resistance to PI3K/mTOR pathway inhibition was associated with the expansion of LMS CSCs. Interestingly, we found that EZH2 inhibition, a catalytic component of polycomb repressive complex which plays a critical role in stem cell maintenance, restored sensitivity to PI3K/mTOR pathway inhibition. Importantly, we confirmed the clinical relevance of our findings by analyzing tumor samples from patients who showed secondary resistance after treatment with a PI3Kα inhibitor.
CONCLUSIONS
Altogether, our findings suggest that CSCs have a strong impact on the outcome of patients with LMS and that combining PI3K/mTOR and EZH2 inhibitors may represent a promising strategy in this setting.

Identifiants

pubmed: 30683135
doi: 10.1186/s13045-018-0694-1
pii: 10.1186/s13045-018-0694-1
pmc: PMC6347793
doi:

Substances chimiques

Imidazoles 0
Phosphoinositide-3 Kinase Inhibitors 0
Protein Kinase Inhibitors 0
Quinolines 0
Aldehyde Dehydrogenase 1 Family EC 1.2.1
EZH2 protein, human EC 2.1.1.43
Enhancer of Zeste Homolog 2 Protein EC 2.1.1.43
MTOR protein, human EC 2.7.1.1
TOR Serine-Threonine Kinases EC 2.7.11.1
dactolisib RUJ6Z9Y0DT

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

11

Commentaires et corrections

Type : ExpressionOfConcernIn

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Auteurs

Benjamin Fourneaux (B)

Université de Bordeaux, Bordeaux, France.
Institut National de la Santé et de la Recherche Medicale (INSERM) U1218, Institut Bergonié, 229 Cours de l'Argonne, 33000, Bordeaux, France.

Aurélien Bourdon (A)

Institut National de la Santé et de la Recherche Medicale (INSERM) U1218, Institut Bergonié, 229 Cours de l'Argonne, 33000, Bordeaux, France.

Bérengère Dadone (B)

Department of Pathology, Nice University Hospital, Nice, France.

Carlo Lucchesi (C)

Institut National de la Santé et de la Recherche Medicale (INSERM) U1218, Institut Bergonié, 229 Cours de l'Argonne, 33000, Bordeaux, France.

Scott R Daigle (SR)

Epizyme, Inc., Cambridge, MA, USA.

Elodie Richard (E)

Université de Bordeaux, Bordeaux, France.
Institut National de la Santé et de la Recherche Medicale (INSERM) U1218, Institut Bergonié, 229 Cours de l'Argonne, 33000, Bordeaux, France.

Audrey Laroche-Clary (A)

Université de Bordeaux, Bordeaux, France.
Institut National de la Santé et de la Recherche Medicale (INSERM) U1218, Institut Bergonié, 229 Cours de l'Argonne, 33000, Bordeaux, France.

François Le Loarer (F)

Department of Medical Oncology, Institut Bergonié, Bordeaux, France.

Antoine Italiano (A)

Institut National de la Santé et de la Recherche Medicale (INSERM) U1218, Institut Bergonié, 229 Cours de l'Argonne, 33000, Bordeaux, France. a.italiano@bordeaux.unicancer.fr.
Department of Medical Oncology, Institut Bergonié, Bordeaux, France. a.italiano@bordeaux.unicancer.fr.

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Classifications MeSH