EZH2 is a potential therapeutic target for H3K27M-mutant pediatric gliomas


Por: Mohammad F, Weissmann S, Leblanc B, Pandey DP, Højfeldt JW, Comet I, Zheng C, Johansen JV, Rapin N, Porse BT, Tvardovskiy A, Jensen ON, Gene-Olaciregui N, Lavarino C, Suñol M, de Torres C, Mora J, Carcaboso AM and Helin K

Publicada: 1 abr 2017 Ahead of Print: 27 feb 2017
Resumen:
Diffuse intrinsic pontine glioma (DIPG) is an aggressive brain tumor that is located in the pons and primarily affects children. Nearly 80% of DIPGs harbor mutations in histone H3 genes, wherein lysine 27 is substituted with methionine (H3K27M). H3K27M has been shown to inhibit polycomb repressive complex 2 (PRC2), a multiprotein complex responsible for the methylation of H3 at lysine 27 (H3K27me), by binding to its catalytic subunit EZH2. Although DIPGs with the H3K27M mutation show global loss of H3K27me3, several genes retain H3K27me3. Here we describe a mouse model of DIPG in which H3K27M potentiates tumorigenesis. Using this model and primary patient-derived DIPG cell lines, we show that H3K27M-expressing tumors require PRC2 for proliferation. Furthermore, we demonstrate that small-molecule EZH2 inhibitors abolish tumor cell growth through a mechanism that is dependent on the induction of the tumor-suppressor protein p16INK4A. Genome-wide enrichment analyses show that the genes that retain H3K27me3 in H3K27M cells are strong polycomb targets. Furthermore, we find a highly significant overlap between genes that retain H3K27me3 in the DIPG mouse model and in human primary DIPGs expressing H3K27M. Taken together, these results show that residual PRC2 activity is required for the proliferation of H3K27M-expressing DIPGs, and that inhibition of EZH2 is a potential therapeutic strategy for the treatment of these tumors.

Filiaciones:
Mohammad F:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

Weissmann S:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

Leblanc B:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

 The Danish Stem Cell Center (Danstem), University of Copenhagen, Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark

Pandey DP:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

Højfeldt JW:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

Comet I:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

Zheng C:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

Johansen JV:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

Rapin N:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 The Danish Stem Cell Center (Danstem), University of Copenhagen, Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark

 The Finsen Laboratory, Rigshospitalet, Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark

 The Bioinformatics Centre, Department of Biology, University of Copenhagen, Copenhagen, Denmark

Porse BT:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 The Danish Stem Cell Center (Danstem), University of Copenhagen, Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark

 The Finsen Laboratory, Rigshospitalet, Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark

Tvardovskiy A:
 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

 Department of Biochemistry and Molecular Biology, VILLUM Center for Bioanalytical Sciences, University of Southern Denmark, Odense M, Denmark

Jensen ON:
 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

 Department of Biochemistry and Molecular Biology, VILLUM Center for Bioanalytical Sciences, University of Southern Denmark, Odense M, Denmark

Lavarino C:
 Department of Pediatric Hematology and Oncology, Hospital Sant Joan de Déu Barcelona, Barcelona, Spain

Suñol M:
 Pathology, Hospital Sant Joan de Déu Barcelona, Barcelona, Spain

de Torres C:
 Department of Pediatric Hematology and Oncology, Hospital Sant Joan de Déu Barcelona, Barcelona, Spain

Mora J:
 Department of Pediatric Hematology and Oncology, Hospital Sant Joan de Déu Barcelona, Barcelona, Spain

Carcaboso AM:
 Department of Pediatric Hematology and Oncology, Hospital Sant Joan de Déu Barcelona, Barcelona, Spain

Helin K:
 Biotech Research and Innovation Centre (BRIC), University of Copenhagen, Copenhagen, Denmark

 Centre for Epigenetics, University of Copenhagen, Copenhagen, Denmark

 The Danish Stem Cell Center (Danstem), University of Copenhagen, Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark
ISSN: 10788956





NATURE MEDICINE
Editorial
NATURE PORTFOLIO, HEIDELBERGER PLATZ 3, BERLIN 14197, GERMANY, Reino Unido
Tipo de documento: Article
Volumen: 23 Número: 4
Páginas: 483-492
WOS Id: 000398768100016
ID de PubMed: 28263309

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