TERT promoter wild-type glioblastomas show distinct clinical features and frequent PI3K pathway mutations

Acta Neuropathol Commun. 2018 Oct 17;6(1):106. doi: 10.1186/s40478-018-0613-2.

Abstract

TERT promoter (TERTp) mutations are found in the majority of World Health Organization (WHO) grade IV adult IDH wild-type glioblastoma (IDH-wt GBM). Here, we characterized the subset of IDH-wt GBMs that do not have TERTp mutations. In a cohort of 121 adult grade IV gliomas, we identified 109 IDH-wt GBMs, after excluding 11 IDH-mutant cases and one H3F3A -mutant case. Within the IDH-wt cases, 16 cases (14.7%) were TERTp wild-type (TERTp-wt). None of the 16 had BRAF V600E or H3F3A G34 hotspot mutations. When compared to TERTp mutants, patients with TERTp-wt GBMs, were significantly younger at first diagnosis (53.2 years vs. 60.7 years, p = 0.0096), and were more frequently found to have cerebellar location (p = 0.0027). Notably, 9 of 16 (56%) of TERTp-wt GBMs contained a PIK3CA or PIK3R1 mutation, while only 16/93 (17%) of TERTp-mutant GBMs harbored these alterations (p = 0.0018). As expected, 8/16 (50%) of TERTp-wt GBMs harbored mutations in the BAF complex gene family (ATRX, SMARCA4, SMARCB1, and ARID1A), compared with only 8/93 (9%) of TERTp-mutant GBMs (p = 0.0003). Mutations in BAF complex and PI3K pathway genes co-occurred more frequently in TERTp-wt GBMs (p = 0.0002), an association that has been observed in other cancers, suggesting a functional interaction indicative of a distinct pathway of gliomagenesis. Overall, our finding highlights heterogeneity within WHO-defined IDH wild-type GBMs and enrichment of the TERTp-wt subset for BAF/PI3K-altered tumors, potentially comprising a distinct clinical subtype of gliomas.

Keywords: BAF complex; Cerebellum; Glioma; H3F3A; IDH1; PI3K pathway; TERT promoter.

Publication types

  • Multicenter Study
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Brain Neoplasms / genetics*
  • Brain Neoplasms / metabolism
  • Cohort Studies
  • DNA Modification Methylases / metabolism
  • DNA Repair Enzymes / metabolism
  • Female
  • Gene Expression Regulation, Neoplastic / genetics
  • Glioblastoma / genetics*
  • Glioblastoma / metabolism
  • Humans
  • Isocitrate Dehydrogenase / genetics
  • Male
  • Middle Aged
  • Mutation / genetics*
  • Phosphatidylinositol 3-Kinases / genetics*
  • Promoter Regions, Genetic / genetics*
  • Signal Transduction / genetics
  • Telomerase / genetics*
  • Tumor Suppressor Proteins / metabolism
  • Young Adult

Substances

  • Tumor Suppressor Proteins
  • Isocitrate Dehydrogenase
  • IDH1 protein, human
  • DNA Modification Methylases
  • MGMT protein, human
  • Phosphatidylinositol 3-Kinases
  • TERT protein, human
  • Telomerase
  • DNA Repair Enzymes