Central mucoepidermoid carcinoma arising from glandular odontogenic cyst confirmed by analysis of MAML2 rearrangement: A case report |
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| Authors: | 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 |
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| Affiliation: | 1. Department of Oral and Maxillofacial Pathobiology, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, Japan;2. Center of Oral Clinical Examination, Hiroshima University Hospital, Hiroshima, Japan;3. Department of Pathology, Cancer Institute Hospital, Japanese Foundation for Cancer Research, Tokyo, Japan;4. Pathology Project for Molecular Targets, Cancer Institute Hospital, Japanese Foundation for Cancer Research, Tokyo, Japan;5. Department of Molecular Oral Medicine and Maxillofacial Surgery, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, Japan;6. Department of Oral and Maxillofacial Radiology, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, Japan |
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| 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. |
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| Keywords: | Central mucoepidermoid carcinoma CRTC3‐MAML2 Glandular odontogenic cyst |
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