Novel cancer gene discovery using a forward genetic screen in RCAS-PDGFB-driven gliomas

Neuro Oncol. 2023 Jan 5;25(1):97-107. doi: 10.1093/neuonc/noac158.

Abstract

Background: Malignant gliomas, the most common malignant brain tumors in adults, represent a heterogeneous group of diseases with poor prognosis. Retroviruses can cause permanent genetic alterations that modify genes close to the viral integration site.

Methods: Here we describe the use of a high-throughput pipeline coupled to the commonly used tissue-specific retroviral RCAS-TVA mouse tumor model system. Utilizing next-generation sequencing, we show that retroviral integration sites can be reproducibly detected in malignant stem cell lines generated from RCAS-PDGFB-driven glioma biopsies.

Results: A large fraction of common integration sites contained genes that have been dysregulated or misexpressed in glioma. Others overlapped with loci identified in previous glioma-related forward genetic screens, but several novel putative cancer-causing genes were also found. Integrating retroviral tagging and clinical data, Ppfibp1 was highlighted as a frequently tagged novel glioma-causing gene. Retroviral integrations into the locus resulted in Ppfibp1 upregulation, and Ppfibp1-tagged cells generated tumors with shorter latency on orthotopic transplantation. In human gliomas, increased PPFIBP1 expression was significantly linked to poor prognosis and PDGF treatment resistance.

Conclusions: Altogether, the current study has demonstrated a novel approach to tagging glioma genes via forward genetics, validating previous results, and identifying PPFIBP1 as a putative oncogene in gliomagenesis.

Keywords: PDGFB; RCAS; forward genetics screen; glioblastoma; liprin-beta-1.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Brain Neoplasms* / pathology
  • Genetic Association Studies
  • Glioma* / pathology
  • Humans
  • Mice
  • Oncogenes
  • Proto-Oncogene Proteins c-sis / genetics

Substances

  • Proto-Oncogene Proteins c-sis
  • PPFIBP1 protein, human