GOPC-ROS1 mosaicism in agminated Spitz naevi: report of two cases

Virchows Arch. 2021 Sep;479(3):559-564. doi: 10.1007/s00428-020-02992-5. Epub 2021 Mar 17.

Abstract

Spitz tumors are genetically associated with activating HRAS point mutations or fusions of either ALK, ROS1, NTRK1, NTRK3, RET, MET, MERTK, LCK, BRAF, MAP3K8, or MAP3K3. All these driver gene alterations are mutually exclusive. We report two cases of agminated Spitz naevi with a GOPC-ROS1 fusion. Both cases occurred on the lower limb of young adults. Since adolescence, pigmented or pink-colored papules have been periodically arising in a limited area of skin. In one case, an ill-defined hyperpigmented macule known since childhood was present in the background. Morphologically, at least five lesions were analyzed from each patient. In one case, all were predominantly junctional pigmented Spitz naevi, and in the other case, all were compound unpigmented Spitz naevi. No atypical features were present. RNA-sequencing revealed a GOPC-ROS1 gene translocation in both cases. Split signals of ROS1 gene in fluorescence in situ hybridization were observed not only in the nests of spitzoid melanocytes but also in the bland basal melanocytes surrounding the proliferations. These findings suggest the presence of a GOPC-ROS1 mosaicism in melanocytes with further emergence of agminated Spitz naevi potentially triggered by other genetic alterations. This expands the spectrum of genetic anomalies described in agminated Spitz naevi and our understanding of the mechanisms involved in their emergence.

Keywords: Agminated Spitz naevi; GOPC; Gene fusion; Mosaicism; ROS1; Spitz naevus.

MeSH terms

  • Adaptor Proteins, Signal Transducing / genetics*
  • Biomarkers, Tumor / genetics*
  • Exome Sequencing
  • Female
  • Gene Fusion*
  • Genetic Predisposition to Disease
  • Golgi Matrix Proteins / genetics*
  • Humans
  • In Situ Hybridization, Fluorescence
  • Male
  • Melanocytes / pathology
  • Mosaicism*
  • Nevus, Epithelioid and Spindle Cell / genetics*
  • Nevus, Epithelioid and Spindle Cell / pathology
  • Phenotype
  • Protein-Tyrosine Kinases / genetics*
  • Proto-Oncogene Proteins / genetics*
  • Sequence Analysis, RNA
  • Skin Neoplasms / genetics*
  • Skin Neoplasms / pathology
  • Young Adult

Substances

  • Adaptor Proteins, Signal Transducing
  • Biomarkers, Tumor
  • GOPC protein, human
  • Golgi Matrix Proteins
  • Proto-Oncogene Proteins
  • Protein-Tyrosine Kinases
  • ROS1 protein, human