Intracranial Ewing sarcoma with whole genome study

Childs Nerv Syst. 2019 Mar;35(3):547-552. doi: 10.1007/s00381-018-3997-1. Epub 2018 Nov 7.

Abstract

Introduction: Ewing sarcoma (ES) as a primary intracranial tumor is very rare. Recently, CNS embryonal tumors with ES-like genomic change have been reported. Patients and methods We report a case of intracranial Ewing sarcoma in a 13-year-old girl who complained of headache and migraine. The tumor had developed in the right middle cranial fossa with a mass effect on the brain with impending transuncal herniation.

Results: Undifferentiated small round cell morphology with completely negative results for friend leukemia integration 1 transcription factor (Fli-1) and a nonspecific cytoplasmic CD99-positive staining pattern mislead the diagnosis as central nervous system (CNS) embryonal tumor, NOS. However, whole genome sequencing (WGS) revealed Ewing sarcoma (EWS)-Fli-1 gene fusion, which was confirmed by fluorescence in situ hybridization study and the diagnosis was revised to ES.

Conclusions: This case is a true intracranial but extra-axial ES confirmed by WGS. We report this case of intracranial ES to demonstrate the importance of marker gene studies using FISH or NGS.

Keywords: CNS embryonal tumor; EWSR1; Ewing sarcoma; Genetics; Next-generation sequencing; Whole genome sequencing.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Cranial Fossa, Middle / pathology
  • Female
  • Humans
  • Oncogene Proteins, Fusion / genetics
  • Proto-Oncogene Protein c-fli-1 / genetics
  • RNA-Binding Protein EWS / genetics
  • Sarcoma, Ewing / diagnosis*
  • Sarcoma, Ewing / genetics
  • Sarcoma, Ewing / pathology*
  • Skull Base Neoplasms / diagnosis*
  • Skull Base Neoplasms / genetics
  • Skull Base Neoplasms / pathology*
  • Whole Genome Sequencing

Substances

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