Genetic Heterogeneity in Cellular Angiofibromas

Genes Chromosomes Cancer. 2024 Aug;63(8):e23262. doi: 10.1002/gcc.23262.

Abstract

Background: Cellular angiofibroma, a rare benign mesenchymal neoplasm, is classified within the 13q/RB1 family of tumors due to morphological, immunohistochemical, and genetic similarities with spindle cell lipoma. Here, genetic data reveal pathogenetic heterogeneity in cellular angiofibroma.

Methods: Three cellular angiofibromas were studied using G-banding/Karyotyping, array comparative genomic hybridization, RNA sequencing, and direct cycling sequencing.

Results: The first tumor carried a del(13)(q12) together with heterozygous loss and minimal expression of the RB1 gene. Tumors two and three displayed chromosome 8 abnormalities associated with chimeras of the pleomorphic adenoma gene 1 (PLAG1). In tumor 2, the cathepsin B (CTSB) fused to PLAG1 (CTSB::PLAG1) while in tumor 3, the mir-99a-let-7c cluster host gene (MIR99AHG) fused to PLAG1 (MIR99AHG::PLAG1), both leading to elevated expression of PLAG1 and insulin growth factor 2.

Conclusion: This study uncovers two genetic pathways contributing to the pathogenetic heterogeneity within cellular angiofibromas. The first aligns with the 13q/RB1 family of tumors and the second involves PLAG1-chimeras. These findings highlight the diverse genetic landscape of cellular angiofibromas, providing insights into potential diagnostic strategies.

Keywords: CTSB::PLAG1; MIR99AHG::PLAG1; 13q/RB1 family of tumors; cathepsin B (CTSB); cellular angiofibroma; genetic heterogeneity; mir‐99a‐let‐7c cluster host gene (MIR99AHG); pleomorphic adenoma gene 1 (PLAG1).

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Angiofibroma* / genetics
  • Angiofibroma* / pathology
  • Cathepsin B
  • Chromosomes, Human, Pair 13* / genetics
  • Chromosomes, Human, Pair 8 / genetics
  • Comparative Genomic Hybridization
  • DNA-Binding Proteins / genetics
  • Female
  • Genetic Heterogeneity*
  • Humans
  • Male
  • MicroRNAs / genetics
  • Middle Aged
  • Retinoblastoma Binding Proteins / genetics
  • Ubiquitin-Protein Ligases / genetics

Substances

  • PLAG1 protein, human
  • DNA-Binding Proteins
  • RB1 protein, human
  • Retinoblastoma Binding Proteins
  • MicroRNAs
  • Ubiquitin-Protein Ligases
  • CTSB protein, human
  • mirnlet7 microRNA, human
  • Cathepsin B