SS18-SSX drives CREB activation in synovial sarcoma.


Journal

Cellular oncology (Dordrecht)
ISSN: 2211-3436
Titre abrégé: Cell Oncol (Dordr)
Pays: Netherlands
ID NLM: 101552938

Informations de publication

Date de publication:
Jun 2022
Historique:
accepted: 08 04 2022
pubmed: 14 5 2022
medline: 15 6 2022
entrez: 13 5 2022
Statut: ppublish

Résumé

Synovial sarcoma (SySa) is a rare soft tissue tumor characterized by a reciprocal t(X;18) translocation. The chimeric SS18-SSX fusion protein represents the major driver of the disease, acting as aberrant transcriptional dysregulator. Oncogenic mechanisms whereby SS18-SSX mediates sarcomagenesis are incompletely understood, and strategies to selectively target SySa cells remain elusive. Based on results of Phospho-Kinase screening arrays, we here investigate the functional and therapeutic relevance of the transcription factor CREB in SySa tumorigenesis. Immunohistochemistry of phosphorylated CREB and its downstream targets (Rb, Cyclin D1, PCNA, Bcl-xL and Bcl-2) was performed in a large cohort of SySa. Functional aspects of CREB activity, including SS18-SSX driven circuits involved in CREB activation, were analyzed in vitro employing five SySa cell lines and a mesenchymal stem cell model. CREB mediated transcriptional activity was modulated by RNAi-mediated knockdown and small molecule inhibitors (666-15, KG-501, NASTRp and Ro 31-8220). Anti-proliferative effects of the CREB inhibitor 666-15 were tested in SySa avian chorioallantoic membrane and murine xenograft models in vivo. We show that CREB is phosphorylated and activated in SySa, accompanied by downstream target expression. Human mesenchymal stem cells engineered to express SS18-SSX promote CREB expression and phosphorylation. Conversely, RNAi-mediated knockdown of SS18-SSX impairs CREB phosphorylation in SySa cells. Inhibition of CREB activity reduces downstream target expression, accompanied by suppression of SySa cell proliferation and induction of apoptosis in vitro and in vivo. In conclusion, our data underline an essential role of CREB in SySa tumorigenesis and provides evidence for molecular targeted therapies.

Identifiants

pubmed: 35556229
doi: 10.1007/s13402-022-00673-w
pii: 10.1007/s13402-022-00673-w
pmc: PMC9187574
doi:

Substances chimiques

Oncogene Proteins, Fusion 0

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

399-413

Subventions

Organisme : deutsche forschungsgemeinschaft
ID : HA4441/2-1
Organisme : wilhelm sander-stiftung
ID : 2016.099.1
Organisme : medizinische fakultät, westfälische wilhelms-universität münster
ID : HU121421
Organisme : medizinische fakultät, westfälische wilhelms-universität münster
ID : TR121716
Organisme : medizinische fakultät, westfälische wilhelms-universität münster
ID : TR122011

Informations de copyright

© 2022. The Author(s).

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Auteurs

Magdalene Cyra (M)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Miriam Schulte (M)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Ruth Berthold (R)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Lorena Heinst (L)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Esther-Pia Jansen (EP)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Inga Grünewald (I)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Sandra Elges (S)

Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Olle Larsson (O)

Departments of Oncology and Pathology, The Karolinska Institute, Stockholm, Sweden.

Christoph Schliemann (C)

Department of Medicine A, Hematology, Oncology and Respiratory Medicine, Münster University Hospital, Münster, Germany.

Konrad Steinestel (K)

Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Institute of Pathology and Molecular Pathology, Bundeswehrkrankenhaus Ulm, Ulm, Germany.

Susanne Hafner (S)

Institute of Pharmacology of Natural Products and Clinical Pharmacology, Ulm University, Ulm, Germany.

Thomas Simmet (T)

Institute of Pharmacology of Natural Products and Clinical Pharmacology, Ulm University, Ulm, Germany.

Eva Wardelmann (E)

Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Sareetha Kailayangiri (S)

Department of Pediatric Hematology and Oncology, University Children's Hospital Münster, Münster, Germany.

Claudia Rossig (C)

Department of Pediatric Hematology and Oncology, University Children's Hospital Münster, Münster, Germany.

Ilka Isfort (I)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany.

Marcel Trautmann (M)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany. marcel.trautmann@ukmuenster.de.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany. marcel.trautmann@ukmuenster.de.

Wolfgang Hartmann (W)

Division of Translational Pathology, Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany. wolfgang.hartmann@ukmuenster.de.
Gerhard-Domagk-Institute of Pathology, Münster University Hospital, Münster, Germany. wolfgang.hartmann@ukmuenster.de.

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