Central mucoepidermoid carcinoma arising from glandular odontogenic cyst confirmed by analysis of MAML2 rearrangement: A case report

Atsuhiro Nagasaki, Ikuko Ogawa, Yukiko Sato, Kengo Takeuchi, Masae Kitagawa, Toshinori Ando, Shinnichi Sakamoto, Madhu Shrestha, Kaori Uchisako, Koichi Koizumi, Shigeaki Toratani, Masaru Konishi, Takashi Takata

Research output: Contribution to journalArticlepeer-review

Abstract

Central mucoepidermoid carcinoma (MEC) poses a diagnostic challenge because of its rarity and histological overlap with glandular odontogenic cyst (GOC). In MEC of both salivary glands and jaws, MAML2 arrangement has been well known as the specific gene alteration. We report a case of central MEC arising from GOC diagnosed by MAML2 fusion gene. A 57-year-old male presented a multilocular cystic lesion in left molar region of the mandible. Histopathologically, multiple cysts lined by thin cuboidal or non-keratinized squamous epithelium with small duct-like structures, mucous cells and ciliated cells were present. It was diagnosed as GOC. The recurrent lesion after nine years showed the proliferation of many cystic and solid nests composed of epidermoid, mucous and intermediated cells. Nested PCR revealed CRTC3-MAML2 fusion gene in the recurrent lesion, but not in the primary one. Similarly, MAML-2 rearrangement by FISH analysis was positive in the recurrent lesion, while negative for the primary one, thus confirming the diagnosis of central MEC arising from GOC. Analysis of MAML2 rearrangement can be used as a supportive evidence to distinguish central MEC from GOC.

Original languageEnglish
Pages (from-to)31-35
Number of pages5
JournalPathology international
Volume68
Issue number1
DOIs
Publication statusPublished - 2018 Jan
Externally publishedYes

Keywords

  • Central mucoepidermoid carcinoma
  • CRTC3-MAML2
  • Glandular odontogenic cyst

ASJC Scopus subject areas

  • Pathology and Forensic Medicine

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