Abstract
Diffuse intrinsic pontine glioma (DIPG) is an incurable tumor that arises in the brainstem of children. To date there is not a single approved drug to effectively treat these tumors and thus novel therapies are desperately needed. Recent studies suggest that a significant fraction of these tumors contain alterations in cell cycle regulatory genes including amplification of the D-type cyclins and CDK4/6, and less commonly, loss of Ink4a-ARF leading to aberrant cell proliferation. In this study, we evaluated the therapeutic approach of targeting the cyclin-CDK-Retinoblastoma (Rb) pathway in a genetically engineered PDGF-B-driven brainstem glioma (BSG) mouse model. We found that PD-0332991 (PD), a CDK4/6 inhibitor, induces cell-cycle arrest in our PDGF-B; Ink4a-ARF deficient model both in vitro and in vivo. By contrast, the PDGF-B; p53 deficient model was mostly resistant to treatment with PD. We noted that a 7-day treatment course with PD significantly prolonged survival by 12% in the PDGF-B; Ink4a-ARF deficient BSG model. Furthermore, a single dose of 10 Gy radiation therapy (RT) followed by 7 days of treatment with PD increased the survival by 19% in comparison to RT alone. These findings provide the rationale for evaluating PD in children with Ink4a-ARF deficient gliomas.
MeSH Terms
Animals
Antineoplastic Agents/pharmacology
Brain Stem Neoplasms/genetics,mortality,pathology,therapy
Cell Cycle Checkpoints/drug effects,radiation effects
Cell Proliferation/drug effects,radiation effects
Cyclin-Dependent Kinase 4/antagonists & inhibitors,genetics,metabolism
Cyclin-Dependent Kinase 6/antagonists & inhibitors,genetics,metabolism
Cyclin-Dependent Kinase Inhibitor p16/deficiency,genetics
Disease Models, Animal
Drug Administration Schedule
Gamma Rays
Gene Expression Regulation, Neoplastic/drug effects,radiation effects
Glioma/genetics,mortality,pathology,therapy
Mice
Oncogene Proteins, Fusion/deficiency,genetics
Piperazines/pharmacology
Proto-Oncogene Proteins c-sis/genetics,metabolism
Pyridines/pharmacology
Retinoblastoma Protein/genetics,metabolism
Signal Transduction
Survival Analysis
Chemicals
Antineoplastic Agents
Cdkn2a protein, mouse
Cyclin-Dependent Kinase Inhibitor p16
Oncogene Proteins, Fusion
Piperazines
Proto-Oncogene Proteins c-sis
Pyridines
Retinoblastoma Protein
Cdk4 protein, mouse
Cdk6 protein, mouse
Cyclin-Dependent Kinase 4
Cyclin-Dependent Kinase 6
palbociclib
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Barton Kelly L
Department of Pediatrics, Duke University, Durham, North Carolina, United States of America ; Preston Robert Tisch Brain Tumor Center, Duke University, Durham, North Carolina, United States of America.
Misuraca Katherine
Cordero Francisco
Dobrikova Elena
Min Hooney D
Gromeier Matthias
Kirsch David G
Becher Oren J
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