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Case Report
Satoshi Maruyama
Niigata University Medical and Dental Hospital
Corresponding Author
ORCiD: https://orcid.org/0000-0001-9036-1536
License:
This work is licensed under a CC BY 4.0 License. Read Full License
Background
Central 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 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 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.
Conclusions
Our 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.
Keywords
central mucoepidermoid carcinoma, glandular odontogenic cyst, immunohistochemistry, cytokeratin 13, MAML-2
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CURRENT STATUS: Published
View the published article at Diagnostic Pathology.
Version 1
Posted 29 Jan, 2021
No community comments so far
Editorial decision: Major Revision
On 06 Apr, 2021
Reviewer #2 agreed
On 03 Apr, 2021
Review #2 received
Received 03 Apr, 2021
Review #1 received
Received 14 Mar, 2021
Reviewer #1 agreed
On 12 Mar, 2021
Reviewers invited
Invitations sent on 09 Mar, 2021
Editor assigned
On 19 Jan, 2021
Submission checks complete
On 19 Jan, 2021
Editor invited
On 19 Jan, 2021
First submitted
On 18 Jan, 2021
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Subject Areas
Pathology
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A new preprint design is coming!
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See the published version of this preprint at Diagnostic Pathology.
This is a preprint, a preliminary version of a manuscript that has not completed peer review at a journal. Research Square does not conduct peer review prior to posting preprints. The posting of a preprint on this server should not be interpreted as an endorsement of its validity or suitability for dissemination as established information or for guiding clinical practice.
Learn more about In Review
Case Report
Satoshi Maruyama
Niigata University Medical and Dental Hospital
Corresponding Author
ORCiD: https://orcid.org/0000-0001-9036-1536
Badges
Prescreen
This manuscript passed our Prescreen assessment
Complete author information
Appropriate declaration statements
Potential risks to human health
Prescreen
Peer Review Timeline
CURRENT STATUS: Published
View the published article at Diagnostic Pathology.
Version 1
Posted 29 Jan, 2021
No community comments so far
Editorial decision: Major Revision
On 06 Apr, 2021
Reviewer #2 agreed
On 03 Apr, 2021
Review #2 received
Received 03 Apr, 2021
Review #1 received
Received 14 Mar, 2021
Reviewer #1 agreed
On 12 Mar, 2021
Reviewers invited
Invitations sent on 09 Mar, 2021
Editor assigned
On 19 Jan, 2021
Submission checks complete
On 19 Jan, 2021
Editor invited
On 19 Jan, 2021
First submitted
On 18 Jan, 2021
Metrics
Comments: 0
PDF Downloads: ...
HTML Views: ...
Subject Areas
Pathology
License:
This work is licensed under a CC BY 4.0 License. Read Full License
View the published article at Diagnostic Pathology.
Background
Central 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 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 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.
Conclusions
Our 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.
Figure 1
Figure 2
Figure 3
Figure 4
Figure 5
This is a list of supplementary files associated with this preprint. Click to download.
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