Common progenitor origin for Rosai-Dorfman disease and clear cell sarcoma

J Pathol. 2024 Nov;264(3):243-249. doi: 10.1002/path.6345. Epub 2024 Sep 3.

Abstract

Histiocytic neoplasms (HNs) in adults have been reported to be associated with a high prevalence of coexisting haematological and solid malignancies. While a proportion of coexisting HNs and haematological malignancies share identical genetic alterations, the genetic association between HNs and solid malignancies has scarcely been reported. We report a case of Rosai-Dorfman disease (RDD) complicated by coexisting clear cell sarcoma (CCS). RDD is a rare HN. CCS is an ultrarare soft tissue sarcoma with a poor prognosis. Mutation analysis with whole-exome sequencing revealed six shared somatic alterations including NRAS p.G12S and TP53 c.559+1G>A in both the RDD and CCS tissue. This is the first evidence of a clonal relationship between RDD and solid malignancies using mutational analysis. We hypothesise that neural crest cells, which originate in CCS, are likely the common cells of origin for RDD and CCS. This case helps to unravel the underlying clinicopathological mechanisms of increased association of solid malignancies in HNs. © 2024 The Author(s). The Journal of Pathology published by John Wiley & Sons Ltd on behalf of The Pathological Society of Great Britain and Ireland.

Keywords: NRAS mutation; Rosai–Dorfman disease; TP53 mutation; clear cell sarcoma; histiocytic neoplasms; neural crest cells; whole‐exome sequencing.

Publication types

  • Case Reports

MeSH terms

  • DNA Mutational Analysis
  • Exome Sequencing
  • Female
  • GTP Phosphohydrolases
  • Histiocytosis, Sinus* / genetics
  • Histiocytosis, Sinus* / pathology
  • Humans
  • Male
  • Membrane Proteins
  • Middle Aged
  • Mutation*
  • Neoplasms, Multiple Primary / genetics
  • Neoplasms, Multiple Primary / pathology
  • Sarcoma, Clear Cell* / genetics
  • Sarcoma, Clear Cell* / pathology
  • Tumor Suppressor Protein p53 / genetics

Substances

  • NRAS protein, human
  • Tumor Suppressor Protein p53
  • Membrane Proteins
  • GTP Phosphohydrolases