Epithelioid variant of gastrointestinal stromal tumor harboring PDGFRA mutation and MLH1 gene alteration: A case report

Pathol Int. 2019 Sep;69(9):541-546. doi: 10.1111/pin.12830. Epub 2019 Jul 4.

Abstract

Gastrointestinal stromal tumors (GISTs) are the most important and common mesenchymal tumors of the gastrointestinal tract, especially in the stomach. GISTs are usually driven by activating mutations in either KIT or PDGFRA genes. It is known that activating gene mutations predicts, to a certain extent, not only the morphology of the tumor cells but also a response to treatment with tyrosine kinase inhibitors. Here, we present a case of an epithelioid variant of GIST harboring PDGFRA and MLH1 gene alterations in the stomach of a 55-year-old Japanese woman. The tumor of 98 mm with multiple cysts showed exophytic growth from the gastric fundus. Histopathologically, it consisted of scattered medium-sized epithelioid tumor cells in a loose myxoid background. Based on c-kit and DOG-1 immunoreactivity and a PDGFRA mutation (p.Trp559_Arg560del), the tumor was diagnosed as an epithelioid variant GIST. Interestingly, it had a gene alteration (p.Met524Ile) in the MLH1 gene of unknown pathogenicity. It was assigned to Group 3a (low risk for malignant behavior). After surgery, the patient has been on imatinib therapy and disease-free for 10 months.

Keywords: GIST; MLH1; PDGFRA; epithelioid variant; stomach.

Publication types

  • Case Reports

MeSH terms

  • Amino Acid Substitution
  • Anoctamin-1 / immunology
  • Antineoplastic Agents / therapeutic use*
  • Epithelioid Cells / pathology
  • Exons / genetics
  • Female
  • Gastrointestinal Neoplasms / diagnostic imaging*
  • Gastrointestinal Neoplasms / drug therapy
  • Gastrointestinal Neoplasms / genetics
  • Gastrointestinal Neoplasms / pathology
  • Gastrointestinal Stromal Tumors / diagnostic imaging*
  • Gastrointestinal Stromal Tumors / drug therapy
  • Gastrointestinal Stromal Tumors / genetics
  • Gastrointestinal Stromal Tumors / pathology
  • Gastrointestinal Tract / pathology
  • Humans
  • Imatinib Mesylate / therapeutic use*
  • Middle Aged
  • MutL Protein Homolog 1 / genetics*
  • Mutation
  • Neoplasm Proteins / immunology
  • Proto-Oncogene Proteins c-kit / immunology
  • Receptor, Platelet-Derived Growth Factor alpha / genetics*

Substances

  • ANO1 protein, human
  • Anoctamin-1
  • Antineoplastic Agents
  • MLH1 protein, human
  • Neoplasm Proteins
  • Imatinib Mesylate
  • KIT protein, human
  • Proto-Oncogene Proteins c-kit
  • Receptor, Platelet-Derived Growth Factor alpha
  • MutL Protein Homolog 1