Abstract
Members of the nuclear factor-κB (NF-κB) family of transcriptional regulators are central mediators of the cellular inflammatory response. Although constitutive NF-κB signalling is present in most human tumours, mutations in pathway members are rare, complicating efforts to understand and block aberrant NF-κB activity in cancer. Here we show that more than two-thirds of supratentorial ependymomas contain oncogenic fusions between RELA, the principal effector of canonical NF-κB signalling, and an uncharacterized gene, C11orf95. In each case, C11orf95-RELA fusions resulted from chromothripsis involving chromosome 11q13.1. C11orf95-RELA fusion proteins translocated spontaneously to the nucleus to activate NF-κB target genes, and rapidly transformed neural stem cells--the cell of origin of ependymoma--to form these tumours in mice. Our data identify a highly recurrent genetic alteration of RELA in human cancer, and the C11orf95-RELA fusion protein as a potential therapeutic target in supratentorial ependymoma.
Publication types
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Research Support, N.I.H., Extramural
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Research Support, Non-U.S. Gov't
MeSH terms
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Adaptor Proteins, Signal Transducing / genetics
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Adaptor Proteins, Signal Transducing / metabolism
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Animals
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Base Sequence
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Brain Neoplasms / genetics
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Brain Neoplasms / metabolism
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Brain Neoplasms / pathology
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Cell Line
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Cell Nucleus / metabolism
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Cell Transformation, Neoplastic* / genetics
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Chromosomes, Human, Pair 11 / genetics
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Ependymoma / genetics*
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Ependymoma / metabolism*
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Ependymoma / pathology
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Female
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Humans
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Mice
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Models, Genetic
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Molecular Sequence Data
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NF-kappa B / genetics
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NF-kappa B / metabolism*
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Neural Stem Cells / metabolism
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Neural Stem Cells / pathology
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Oncogene Proteins, Fusion / genetics
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Oncogene Proteins, Fusion / metabolism
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Phosphoproteins / genetics
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Phosphoproteins / metabolism
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Proteins / genetics
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Proteins / metabolism*
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Signal Transduction*
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Transcription Factor RelA / genetics
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Transcription Factor RelA / metabolism*
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Transcription Factors
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Translocation, Genetic / genetics
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YAP-Signaling Proteins
Substances
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Adaptor Proteins, Signal Transducing
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C11orf95 protein, human
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NF-kappa B
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Oncogene Proteins, Fusion
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Phosphoproteins
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Proteins
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RELA protein, human
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Transcription Factor RelA
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Transcription Factors
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YAP-Signaling Proteins
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YAP1 protein, human