Epigenetic landscape reorganisation and reactivation of embryonic development genes are associated with malignancy in IDH-mutant astrocytoma

Acta Neuropathol. 2024 Oct 9;148(1):50. doi: 10.1007/s00401-024-02811-0.

Abstract

Accurate grading of IDH-mutant gliomas defines patient prognosis and guides the treatment path. Histological grading is challenging, and aside from CDKN2A/B homozygous deletions in IDH-mutant astrocytomas, there are no other objective molecular markers used for grading. RNA-sequencing was conducted on primary IDH-mutant astrocytomas (n = 138) included in the prospective CATNON trial, which was performed to assess the prognostic effect of adjuvant and concurrent temozolomide. We integrated the RNA-sequencing data with matched DNA-methylation and NGS data. We also used multi-omics data from IDH-mutant astrocytomas included in the TCGA dataset and validated results on matched primary and recurrent samples from the GLASS-NL study. Since discrete classes do not adequately capture grading of these tumours, we utilised DNA-methylation profiles to generate a Continuous Grading Coefficient (CGC) based on classification scores from a CNS-tumour classifier. CGC was an independent predictor of survival outperforming current WHO-CNS5 and methylation-based classification. Our RNA-sequencing analysis revealed four distinct transcription clusters that were associated with (i) upregulation of cell cycling genes; (ii) downregulation of glial differentiation genes; (iii) upregulation of embryonic development genes (e.g. HOX, PAX, and TBX) and (iv) upregulation of extracellular matrix genes. The upregulation of embryonic development genes was associated with a specific increase of CpG island methylation near these genes. Higher grade IDH-mutant astrocytomas have DNA-methylation signatures that, on the RNA level, are associated with increased cell cycling, tumour cell de-differentiation and extracellular matrix remodelling. These combined molecular signatures can serve as an objective marker for grading of IDH-mutant astrocytomas.

Keywords: Astrocytoma; DNA methylation; HOX; IDH; Molecular grading; RNA sequencing.

MeSH terms

  • Adult
  • Astrocytoma* / genetics
  • Astrocytoma* / pathology
  • Brain Neoplasms* / genetics
  • Brain Neoplasms* / pathology
  • DNA Methylation* / genetics
  • Embryonic Development / genetics
  • Epigenesis, Genetic* / genetics
  • Female
  • Humans
  • Isocitrate Dehydrogenase* / genetics
  • Male
  • Middle Aged
  • Mutation* / genetics
  • Neoplasm Grading

Substances

  • Isocitrate Dehydrogenase