Pediatric Erythroid Sarcoma Diagnostically Confirmed by Identification of a Recurrent NFIA::CBFA2T3 Fusion

Genes Chromosomes Cancer. 2024 Jun;63(6):e23251. doi: 10.1002/gcc.23251.

Abstract

Erythroid sarcoma (ES) is exceedingly rare in the pediatric population with only a handful of reports of de novo cases, mostly occurring in the central nervous system (CNS) or orbit. It is clinically and pathologically challenging and can masquerade as a nonhematopoietic small round blue cell tumor. Clinical presentation of ES without bone marrow involvement makes diagnosis particularly difficult. We describe a 22-month-old female with ES who presented with a 2-cm mass involving the left parotid region and CNS. The presence of crush/fixation artifact from the initial biopsy made definitive classification of this highly proliferative and malignant neoplasm challenging despite an extensive immunohistochemical workup. Molecular studies including RNA-sequencing revealed a NFIA::CBFA2T3 fusion. This fusion has been identified in several cases of de novo acute erythroid leukemia (AEL) and gene expression analysis comparing this case to other AELs revealed a similar transcriptional profile. Given the diagnostically challenging nature of this tumor, clinical RNA-sequencing was essential for establishing a diagnosis.

Keywords: NFIA::CBFA2T3; RNA‐sequencing; acute erythroid leukemia (AEL); erythroid sarcoma (ES); pure erythroid leukemia (PEL).

Publication types

  • Case Reports

MeSH terms

  • Female
  • Humans
  • Infant
  • NFI Transcription Factors* / genetics
  • Oncogene Proteins, Fusion* / genetics
  • Repressor Proteins*
  • Sarcoma* / diagnosis
  • Sarcoma* / genetics
  • Sarcoma* / pathology

Substances

  • CBFA2T3 protein, human
  • NFI Transcription Factors
  • NFIA protein, human
  • Oncogene Proteins, Fusion
  • Repressor Proteins