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


Journal

Pathology international
ISSN: 1440-1827
Titre abrégé: Pathol Int
Pays: Australia
ID NLM: 9431380

Informations de publication

Date de publication:
Sep 2019
Historique:
received: 29 03 2019
accepted: 28 05 2019
pubmed: 6 7 2019
medline: 16 5 2020
entrez: 6 7 2019
Statut: ppublish

Résumé

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.

Identifiants

pubmed: 31273885
doi: 10.1111/pin.12830
pmc: PMC8194368
mid: NIHMS1703708
doi:

Substances chimiques

ANO1 protein, human 0
Anoctamin-1 0
Antineoplastic Agents 0
MLH1 protein, human 0
Neoplasm Proteins 0
Imatinib Mesylate 8A1O1M485B
KIT protein, human EC 2.7.10.1
Proto-Oncogene Proteins c-kit EC 2.7.10.1
Receptor, Platelet-Derived Growth Factor alpha EC 2.7.10.1
MutL Protein Homolog 1 EC 3.6.1.3

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

541-546

Subventions

Organisme : Intramural NIH HHS
ID : ZIA BC011427
Pays : United States

Informations de copyright

© 2019 Japanese Society of Pathology and John Wiley & Sons Australia, Ltd.

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Auteurs

Mizuho Kobayashi (M)

Department of Pathology, Nagoya City West Medical Center, Nagoya, Japan.
Department of Experimental Pathology and Tumor Biology, Graduate School of Medical Sciences, Nagoya City University, Nagoya, Japan.

Shingo Inaguma (S)

Department of Pathology, Aichi Medical University School of Medicine, Nagakute, Japan.

Mark Raffeld (M)

Laboratory of Pathology, National Cancer Institute, Bethesda, USA.

Hiroyuki Kato (H)

Department of Experimental Pathology and Tumor Biology, Graduate School of Medical Sciences, Nagoya City University, Nagoya, Japan.

Shugo Suzuki (S)

Department of Experimental Pathology and Tumor Biology, Graduate School of Medical Sciences, Nagoya City University, Nagoya, Japan.

Takehiro Wakasugi (T)

Gastrointestinal Surgery, Nagoya City West Medical Center, Nagoya, Japan.

Akira Mitsui (A)

Gastrointestinal Surgery, Nagoya City West Medical Center, Nagoya, Japan.

Yoshiyuki Kuwabara (Y)

Gastrointestinal Surgery, Nagoya City West Medical Center, Nagoya, Japan.

Jerzy Lasota (J)

Laboratory of Pathology, National Cancer Institute, Bethesda, USA.

Hiroshi Ikeda (H)

Department of Pathology, Aichi Medical University School of Medicine, Nagakute, Japan.

Markku Miettinen (M)

Laboratory of Pathology, National Cancer Institute, Bethesda, USA.

Satoru Takahashi (S)

Department of Experimental Pathology and Tumor Biology, Graduate School of Medical Sciences, Nagoya City University, Nagoya, Japan.

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Classifications MeSH