Primary Adrenal Angiomatoid Fibrous Histiocytoma With Novel EWSR1-ATF1 Gene Fusion Exon-Exon Breakpoint

Pediatr Dev Pathol. 2019 Oct;22(5):472-474. doi: 10.1177/1093526619830287. Epub 2019 Mar 1.

Abstract

We describe the clinical, pathological, and molecular features of a primary adrenal angiomatoid fibrous histiocytoma (AFH) in an 11-year-old girl presenting with pyrexia of unknown origin. We performed next-generation sequencing-based anchored multiplex polymerase chain reaction (Archer® FusionPlex® sarcoma assay), which revealed an EWSR1-ATF1 gene fusion with novel breakpoints in exon 11 of EWSR1 and exon 3 of ATF1. The pyrexia resolved fully after surgical resection, and the patient was disease-free on follow-up at 1 year and 6 months. This case exemplifies the value of molecular testing of pediatric neoplasms presenting at unusual sites for diagnosis and identification of novel gene fusion breakpoints.

Keywords: Archer FusionPlex sarcoma assay; EWSR1-ATF1 gene fusion; adrenal gland; anchored multiplex polymerase chain reaction; angiomatoid fibrous histiocytoma; pyrexia of unknown origin.

Publication types

  • Case Reports

MeSH terms

  • Adrenal Gland Neoplasms / genetics*
  • Child
  • Exons / genetics
  • Female
  • Histiocytoma, Malignant Fibrous / genetics*
  • Humans
  • Oncogene Proteins, Fusion / genetics*

Substances

  • EWSR1-ATF1 fusion protein, human
  • Oncogene Proteins, Fusion

Supplementary concepts

  • Histiocytoma, Angiomatoid Fibrous