Combined experience of six independent laboratories attempting to create an Ewing sarcoma mouse model

Oncotarget. 2017 May 23;8(21):34141-34163. doi: 10.18632/oncotarget.9388.

Abstract

Ewing sarcoma (ES) involves a tumor-specific chromosomal translocation that produces the EWS-FLI1 protein, which is required for the growth of ES cells both in vitro and in vivo. However, an EWS-FLI1-driven transgenic mouse model is not currently available. Here, we present data from six independent laboratories seeking an alternative approach to express EWS-FLI1 in different murine tissues. We used the Runx2, Col1a2.3, Col1a3.6, Prx1, CAG, Nse, NEFL, Dermo1, P0, Sox9 and Osterix promoters to target EWS-FLI1 or Cre expression. Additional approaches included the induction of an endogenous chromosomal translocation, in utero knock-in, and the injection of Cre-expressing adenovirus to induce EWS-FLI1 expression locally in multiple lineages. Most models resulted in embryonic lethality or developmental defects. EWS-FLI1-induced apoptosis, promoter leakiness, the lack of potential cofactors, and the difficulty of expressing EWS-FLI1 in specific sites were considered the primary reasons for the failed attempts to create a transgenic mouse model of ES.

Keywords: EWS-FLI1; EWS-FLI1 driven transgenic mouse model; Ewing sarcoma.

MeSH terms

  • Adenoviridae / genetics
  • Animals
  • Cell Line, Tumor
  • Disease Models, Animal*
  • Gene Expression Regulation, Neoplastic
  • Gene Knock-In Techniques
  • Humans
  • Mice
  • Mice, Transgenic
  • Neoplasm Transplantation
  • Oncogene Proteins, Fusion / genetics*
  • Promoter Regions, Genetic*
  • Proto-Oncogene Protein c-fli-1 / genetics*
  • RNA-Binding Protein EWS / genetics*
  • Sarcoma, Ewing / genetics
  • Sarcoma, Ewing / pathology*

Substances

  • EWS-FLI fusion protein
  • Oncogene Proteins, Fusion
  • Proto-Oncogene Protein c-fli-1
  • RNA-Binding Protein EWS