Primary malignant ossifying fibromyxoid tumour of the bone. A clinicopathologic and molecular report of two cases

Pathologica. 2020 Dec;112(4):184-190. doi: 10.32074/1591-951X-207. Epub 2020 Nov 3.

Abstract

Objective: To report the exceptional occurrence of ossifying fibromyxoid tumour (OFMT) as a primary bone lesion. OFMT is a rare soft tissue tumour of uncertain differentiation and variable malignant potential, that occurs in adults with a slight male predominance. It is typically located in the subcutis or in the skeletal muscles of the extremities, followed by trunk or head and neck.

Methods: Two cases of OFMT proven to arise from bone are presented. The first is a 65-year old female with a history of rib "osteosarcoma", presenting with an inferior lobe left lung mass. The second is a man with a lytic lesion of the 5th cervical vertebra that recurred shortly after resection. Following H&E and immunohistochemical examination, tumour samples were analysed by NGS and by break-apart FISH to detect rearrangement of the PHF1 and TFE3 genes.

Results: PHF1 gene-rearrangement was identified by FISH on both the primary and the metastatic lesion of first patient. NGS identified a PHF1(intron1) and EPC1 (exon 10) fusion transcript later confirmed by positive PHF1 rearrangement on FISH in the second case.

Conclusions: The demonstration of PHF1 gene rearrangements represents a fundamental ancillary diagnostic test when presented with challenging examples of OFMT.

Keywords: OFMT; PHF1; bone tumours; rare tumours; soft tissue tumours.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Aged
  • Biomarkers, Tumor / analysis
  • Bone Neoplasms* / diagnosis
  • Bone Neoplasms* / genetics
  • Bone Neoplasms* / pathology
  • DNA, Neoplasm / analysis
  • DNA-Binding Proteins / genetics*
  • Female
  • Fibroma, Ossifying* / diagnosis
  • Fibroma, Ossifying* / genetics
  • Fibroma, Ossifying* / pathology
  • Gene Rearrangement
  • High-Throughput Nucleotide Sequencing
  • Humans
  • Immunohistochemistry
  • In Situ Hybridization, Fluorescence
  • Male
  • Neoplasm Recurrence, Local / pathology
  • Polycomb-Group Proteins / genetics*
  • Soft Tissue Neoplasms / diagnosis
  • Soft Tissue Neoplasms / genetics
  • Soft Tissue Neoplasms / pathology
  • Transcription Factors / genetics

Substances

  • Biomarkers, Tumor
  • DNA, Neoplasm
  • DNA-Binding Proteins
  • PHF1 protein, human
  • Polycomb-Group Proteins
  • Transcription Factors