Central Mucoepidermoid Carcinoma Arising Directly From a Glandular Odontogenic Cyst of the Mandible: A Case Report

Abstract BackgroundCentral mucoepidermoid carcinoma (MEC) is a rare salivary gland tumor that affects the jaw bone. Glandular odontogenic cyst (GOC) is also a rare odontogenic developmental cyst with glandular differentiation. GOC shares some histological features with central MEC, and a pre-existing GOC can develop into central MEC. Here, we present a rare case of central MEC developed directly from a pre-existing GOC of the mandible. Case presentationA 67-year-old Japanese man presented with a cystic lesion in the right third molar region. Histologically, the biopsy specimen demonstrated both typical of a GOC component lined with non-keratinized squamous epithelium and a recognizable component of central MEC consisting of polycystic nests with mucous cells, intermediate cells, and epidermoid cells in the cyst wall. The immunohistochemistry for cytokeratin (CK) profile results demonstrated that while both central MEC and GOC expressed CKs 7, 14, 18, and 19, interestingly CK13 was only expressed in GOC. Fluorescence in-situ hybridization (FISH) revealed the rearrangement of the Mastermind like (MAML)-2 gene in both MEC and GOC components. ConclusionsOur case suggests that central MEC and GOC may be in the same spectrum of diseases caused by rearrangement of the MAML-2 gene. At the same time, the expression profile of CK13 was completely different in both central MEC and GOC. This also suggests that central MEC is a distinct tumor from GOC. Thus, we demonstrated the rare case that central MEC may have originated directly from the GOC.

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Central mucoepidermoid carcinoma arising directly from a glandular odontogenic cyst of the mandible: a case report

Diagnostic Pathology ◽

10.1186/s13000-021-01124-0 ◽

2021 ◽

Vol 16 (1) ◽

Author(s):

Satoshi Maruyama ◽

Taisuke Mori ◽

Manabu Yamazaki ◽

Tatsuya Abé ◽

Eijitsu Ryo ◽

...

Keyword(s):

Mucoepidermoid Carcinoma ◽

Rare Case ◽

Salivary Gland Tumor ◽

Third Molar ◽

Odontogenic Cyst ◽

Glandular Odontogenic Cyst ◽

Old Japanese ◽

Central Mucoepidermoid Carcinoma ◽

Intermediate Cells ◽

The Right

Abstract Background Central mucoepidermoid carcinoma (MEC) is a rare salivary gland tumor that affects the jawbone. Glandular odontogenic cyst (GOC) is also a rare odontogenic developmental cyst with glandular differentiation. GOC shares some histological features with central MEC, and a pre-existing GOC can develop into central MEC. Here, we present a rare case of central MEC developed directly from a pre-existing GOC of the mandible. Case presentation A 67-year-old Japanese man presented with a cystic lesion in the right third molar region. Histologically, the biopsy specimen demonstrated both typical findings of a GOC component lined with non-keratinized squamous epithelium and a recognizable component of central MEC consisting of polycystic nests with mucous cells, intermediate cells, and epidermoid cells in the cyst wall. The results from the immunohistochemistry for cytokeratin (CK) profiling demonstrated that, while both central MEC and GOC expressed CKs 7, 14, 18, and 19, CK13 was interestingly exclusively expressed in GOC. Fluorescence in-situ hybridization (FISH) revealed the rearrangement of the Mastermind like (MAML)-2 gene in both the MEC and GOC components. Conclusions Our case suggests that central MEC and GOC may be in the same spectrum of diseases caused by the rearrangement of the MAML-2 gene. However, given that the expression profile of CK13 was completely different between central MEC and GOC, they can be considered as separate tumors. Overall, we demonstrated a rare case in which central MEC may have originated directly from the GOC.

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Glandular Odontogenic Cyst: Review of Literature and Report of a New Case with Cytokeratin-19 Expression

The Open Dentistry Journal ◽

10.2174/1874210601408010001 ◽

2014 ◽

Vol 8 (1) ◽

pp. 1-12 ◽

Cited By ~ 11

Author(s):

Marco Mascitti ◽

Andrea Santarelli ◽

Antonio Sabatucci ◽

Maurizio Procaccini ◽

Lorenzo Lo Muzio ◽

...

Keyword(s):

Mucoepidermoid Carcinoma ◽

Squamous Epithelium ◽

Odontogenic Cyst ◽

Cytokeratin 19 ◽

Review Of Literature ◽

Distinct Entity ◽

Radiographic Findings ◽

Glandular Odontogenic Cyst ◽

Stratified Squamous Epithelium ◽

Central Mucoepidermoid Carcinoma

The glandular odontogenic cyst (GOC) was a rare jawbone cyst described in 1988 as a distinct entity. This lesion can involve either jaw, and the anterior region of the mandible was the most commonly affected area. Clinical and radiographic findings were not specific, and the diagnosis of GOC can be extremely difficult due to the rarity of this lesion. The cyst presented a wall constituted by fibrous connective tissue and was lined by a non-keratinized stratified squamous epithelium of variable thickness. Large areas of the lining epithelium presented cylinder cells, sometimes ciliated. A variable amount of mucina was occasionally noted. Due to the strong similarities, this cyst can be easily misdiag-nosed as a central mucoepidermoid carcinoma (CMEC). Immunohistochemistry may be an aid in diagnosis; in fact has been demonstrated that there were differences in the expression of cytokeratins (CK) in GOC and CMEC. In this study, we reported a new case of GOC in a 38 year female patient. In addition, we carried out a review of 110 previous cases reported in literature.

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

Pathology International ◽

10.1111/pin.12609 ◽

2017 ◽

Vol 68 (1) ◽

pp. 31-35 ◽

Cited By ~ 3

Author(s):

Atsuhiro Nagasaki ◽

Ikuko Ogawa ◽

Yukiko Sato ◽

Kengo Takeuchi ◽

Masae Kitagawa ◽

...

Keyword(s):

Case Report ◽

Mucoepidermoid Carcinoma ◽

Odontogenic Cyst ◽

Glandular Odontogenic Cyst ◽

Central Mucoepidermoid Carcinoma

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Unique case of glandular odontogenic cyst showing mucoepidermoid carcinoma-like islands in cystic wall

BMJ Case Reports ◽

10.1136/bcr-2020-239362 ◽

2021 ◽

Vol 14 (2) ◽

pp. e239362

Author(s):

Harpreet Kaur ◽

Deepika Mishra ◽

Rahul Yadav

Keyword(s):

Mucoepidermoid Carcinoma ◽

Final Diagnosis ◽

Odontogenic Cyst ◽

Odontogenic Cysts ◽

Low Grade ◽

Male Predominance ◽

Glandular Odontogenic Cyst ◽

Jaw Cyst ◽

Intermediate Cells ◽

Anterior Mandible

Glandular odontogenic cyst (GOC) is a very rare jaw cyst accounting for 0.2% of all odontogenic cysts. It presents usually in adults with a slight male predominance. It shows radiological, histopathological and even immunohistochemical overlap with low grade intraosseous mucoepidermoid carcinoma (MEC) but their distinction is crucial. A 57-year-old woman with bilocular radiolucency in the anterior mandible crossing the midline is described here. Microscopy features were consistent with glandular odontogenic cyst but multiple MEC-like islands were seen in the capsule, creating a diagnostic head trip with low grade intraosseous MEC. However, the absence of cellular atypia and epidermoid and intermediate cells led to a final diagnosis of GOC, with close follow-up of the patient recommended. This rare finding shows the relation between GOC and MEC or the origin of MEC from GOC.

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