Literature DB >> 24203892

Activation of mTORC1/mTORC2 signaling in pediatric low-grade glioma and pilocytic astrocytoma reveals mTOR as a therapeutic target.

Marianne Hütt-Cabezas1, Matthias A Karajannis, David Zagzag, Smit Shah, Iren Horkayne-Szakaly, Elisabeth J Rushing, J Douglas Cameron, Deepali Jain, Charles G Eberhart, Eric H Raabe, Fausto J Rodriguez.   

Abstract

BACKGROUND: Previous studies support a role for mitogen-activated protein kinase pathway signaling, and more recently Akt/mammalian target of rapamycin (mTOR), in pediatric low-grade glioma (PLGG), including pilocytic astrocytoma (PA). Here we further evaluate the role of the mTORC1/mTORC2 pathway in order to better direct pharmacologic blockade in these common childhood tumors.
METHODS: We studied 177 PLGGs and PAs using immunohistochemistry and tested the effect of mTOR blockade on 2 PLGG cell lines (Res186 and Res259) in vitro.
RESULTS: Moderate (2+) to strong (3+) immunostaining was observed for pS6 in 107/177 (59%) PAs and other PLGGs, while p4EBP1 was observed in 35/115 (30%), pElF4G in 66/112 (59%), mTOR (total) in 53/113 (47%), RAPTOR (mTORC1 component) in 64/102 (63%), RICTOR (mTORC2 component) in 48/101 (48%), and pAkt (S473) in 63/103 (61%). Complete phosphatase and tensin homolog protein loss was identified in only 7/101 (7%) of cases. In PA of the optic pathways, compared with other anatomic sites, there was increased immunoreactivity for pS6, pElF4G, mTOR (total), RICTOR, and pAkt (P < .05). We also observed increased pS6 (P = .01), p4EBP1 (P = .029), and RICTOR (P = .05) in neurofibromatosis type 1 compared with sporadic tumors. Treatment of the PLGG cell lines Res186 (PA derived) and Res259 (diffuse astrocytoma derived) with the rapalog MK8669 (ridaforolimus) led to decreased mTOR pathway activation and growth.
CONCLUSIONS: These findings suggest that the mTOR pathway is active in PLGG but varies by clinicopathologic subtype. Additionally, our data suggest that mTORC2 is differentially active in optic pathway and neurofibromatosis type 1-associated gliomas. MTOR represents a potential therapeutic target in PLGG that merits further investigation.

Entities:  

Keywords:  mTOR; neurofibromatosis; optic nerve; pediatric glioma; pilocytic astrocytoma

Mesh:

Substances:

Year:  2013        PMID: 24203892      PMCID: PMC3829599          DOI: 10.1093/neuonc/not132

Source DB:  PubMed          Journal:  Neuro Oncol        ISSN: 1522-8517            Impact factor:   12.300


  36 in total

Review 1.  Pediatric low-grade gliomas.

Authors:  Angela J Sievert; Michael J Fisher
Journal:  J Child Neurol       Date:  2009-11       Impact factor: 1.987

2.  Loss of NF1 alleles distinguish sporadic from NF1-associated pilocytic astrocytomas.

Authors:  L Kluwe; C Hagel; M Tatagiba; S Thomas; D Stavrou; H Ostertag; A von Deimling; V F Mautner
Journal:  J Neuropathol Exp Neurol       Date:  2001-09       Impact factor: 3.685

3.  The pharmacology of mTOR inhibition.

Authors:  David A Guertin; David M Sabatini
Journal:  Sci Signal       Date:  2009-04-21       Impact factor: 8.192

4.  Activation of the ERK/MAPK pathway: a signature genetic defect in posterior fossa pilocytic astrocytomas.

Authors:  Tim Forshew; Ruth G Tatevossian; Andrew R J Lawson; Jing Ma; Geoff Neale; Babatunji W Ogunkolade; Tania A Jones; Johan Aarum; James Dalton; Simon Bailey; Tracy Chaplin; Rowena L Carter; Amar Gajjar; Alberto Broniscer; Bryan D Young; David W Ellison; Denise Sheer
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5.  Duplication of 7q34 in pediatric low-grade astrocytomas detected by high-density single-nucleotide polymorphism-based genotype arrays results in a novel BRAF fusion gene.

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6.  Oncogenic RAF1 rearrangement and a novel BRAF mutation as alternatives to KIAA1549:BRAF fusion in activating the MAPK pathway in pilocytic astrocytoma.

Authors:  D T W Jones; S Kocialkowski; L Liu; D M Pearson; K Ichimura; V P Collins
Journal:  Oncogene       Date:  2009-04-13       Impact factor: 9.867

7.  BRAF gene duplication constitutes a mechanism of MAPK pathway activation in low-grade astrocytomas.

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8.  Pilocytic astrocytoma of the optic pathway: a tumour deriving from radial glia cells with a specific gene signature.

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9.  Tandem duplication producing a novel oncogenic BRAF fusion gene defines the majority of pilocytic astrocytomas.

Authors:  David T W Jones; Sylvia Kocialkowski; Lu Liu; Danita M Pearson; L Magnus Bäcklund; Koichi Ichimura; V Peter Collins
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10.  Molecular and phenotypic characterisation of paediatric glioma cell lines as models for preclinical drug development.

Authors:  Dorine A Bax; Suzanne E Little; Nathalie Gaspar; Lara Perryman; Lynley Marshall; Marta Viana-Pereira; Tania A Jones; Richard D Williams; Anita Grigoriadis; Gilles Vassal; Paul Workman; Denise Sheer; Rui M Reis; Andrew D J Pearson; Darren Hargrave; Chris Jones
Journal:  PLoS One       Date:  2009-04-14       Impact factor: 3.240

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  38 in total

Review 1.  Review of low-grade gliomas in children--evolving molecular era and therapeutic insights.

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Journal:  Childs Nerv Syst       Date:  2015-02-27       Impact factor: 1.475

2.  Inhibition of mTORC1 in pediatric low-grade glioma depletes glutathione and therapeutically synergizes with carboplatin.

Authors:  Brad Poore; Ming Yuan; Antje Arnold; Antoinette Price; Jesse Alt; Jeffrey A Rubens; Barbara S Slusher; Charles G Eberhart; Eric H Raabe
Journal:  Neuro Oncol       Date:  2019-02-14       Impact factor: 12.300

3.  Alternative lengthening of telomeres, ATRX loss and H3-K27M mutations in histologically defined pilocytic astrocytoma with anaplasia.

Authors:  Fausto J Rodriguez; Jacqueline A Brosnan-Cashman; Sariah J Allen; M Adelita Vizcaino; Caterina Giannini; Sandra Camelo-Piragua; Milad Webb; Marcus Matsushita; Nitin Wadhwani; Abeer Tabbarah; Dima Hamideh; Liqun Jiang; Liam Chen; Leonidas D Arvanitis; Hussein H Alnajar; John R Barber; Alicia Rodríguez-Velasco; Brent Orr; Christopher M Heaphy
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4.  Screening genes crucial for pediatric pilocytic astrocytoma using weighted gene coexpression network analysis combined with methylation data analysis.

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Review 5.  Pediatric Brain Tumors: Current Knowledge and Therapeutic Opportunities.

Authors:  John Glod; Gilbert J Rahme; Harpreet Kaur; Eric H Raabe; Eugene I Hwang; Mark A Israel
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Review 6.  Targeted therapy in pediatric low-grade glioma.

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7.  Synergistic activity of mTORC1/2 kinase and MEK inhibitors suppresses pediatric low-grade glioma tumorigenicity and vascularity.

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Review 8.  Advances in the management of low-grade gliomas.

Authors:  Amulya A Nageswara Rao; Roger J Packer
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Review 9.  Recent Advances on the Molecular Pathology of Glial Neoplasms in Children and Adults.

Authors:  Fausto J Rodriguez; M Adelita Vizcaino; Ming-Tseh Lin
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10.  mTOR: a new therapeutic target for pediatric low-grade glioma?

Authors:  Fausto J Rodriguez; Eric H Raabe
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