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Squamous cell carcinoma arising within a maxillary odontogenic keratocyst: A rare occurrence

Identifieur interne : 001C84 ( Pmc/Curation ); précédent : 001C83; suivant : 001C85

Squamous cell carcinoma arising within a maxillary odontogenic keratocyst: A rare occurrence

Auteurs : Elnaz Jalali [États-Unis] ; Elie M. Ferneini [États-Unis] ; Kandasamy Rengasamy [États-Unis] ; Aditya Tadinada [États-Unis]

Source :

RBID : PMC:5489670

Abstract

Squamous cell carcinoma (SCC) arising within the lining of an odontogenic keratocyst (OKC) is a rare occurrence. Although potentially locally destructive, OKC is a benign odontogenic process that typically presents with clinical and radiographic features characteristic of a benign intraosseous neoplasm. We present the clinical and radiographic features of a maxillary mass that demonstrated SCC arising from the lining of an OKC. Although the initial clinical and radiographic presentation suggested an infection or malignant neoplasm, biopsies revealed an infiltrative well-differentiated SCC contiguous with and arising from the focus of a pre-existing OKC. The patient subsequently underwent a type II hemi-maxillectomy with neoadjuvant chemoradiation. This report discusses the clinical and radiographic features associated with intraosseous malignancies, especially those arising from an otherwise benign odontogenic lesion. While the majority of OKCs are benign, the current report illustrates the potential for carcinomatous transformation within the lining of an OKC.


Url:
DOI: 10.5624/isd.2017.47.2.135
PubMed: 28680851
PubMed Central: 5489670

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PMC:5489670

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<contrib contrib-type="author">
<name>
<surname>Rengasamy</surname>
<given-names>Kandasamy</given-names>
</name>
<xref ref-type="aff" rid="A3">3</xref>
</contrib>
<contrib contrib-type="author" corresp="yes">
<name>
<surname>Tadinada</surname>
<given-names>Aditya</given-names>
</name>
<xref ref-type="aff" rid="A3">3</xref>
</contrib>
</contrib-group>
<aff id="A1">
<label>1</label>
Eliray Oral and Maxillofacial Radiology Consulting Services, Miami, FL, USA.</aff>
<aff id="A2">
<label>2</label>
Department of Craniofacial Sciences, Division of Oral and Maxillofacial Surgery, University of Connecticut, School of Dental Medicine, Farmington, CT, USA.</aff>
<aff id="A3">
<label>3</label>
Section of Oral and Maxillofacial Radiology, University of Connecticut, School of Dental Medicine, Farmington, CT, USA.</aff>
<author-notes>
<corresp>Correspondence to: Prof. Aditya Tadinada. Section of Oral and Maxillofacial Radiology, University of Connecticut, School of Dental Medicine, 263 Farmington Ave., Farmington, CT 06030, USA. Tel) 1-860-679-2719, Fax) 1-860-679-4760,
<email>Tadinada@uchc.edu</email>
</corresp>
</author-notes>
<pub-date pub-type="ppub">
<month>6</month>
<year>2017</year>
</pub-date>
<pub-date pub-type="epub">
<day>22</day>
<month>6</month>
<year>2017</year>
</pub-date>
<volume>47</volume>
<issue>2</issue>
<fpage>135</fpage>
<lpage>140</lpage>
<history>
<date date-type="received">
<day>10</day>
<month>2</month>
<year>2017</year>
</date>
<date date-type="rev-recd">
<day>07</day>
<month>3</month>
<year>2017</year>
</date>
<date date-type="accepted">
<day>24</day>
<month>3</month>
<year>2017</year>
</date>
</history>
<permissions>
<copyright-statement>Copyright © 2017 by Korean Academy of Oral and Maxillofacial Radiology</copyright-statement>
<copyright-year>2017</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">
<license-p>This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (
<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">http://creativecommons.org/licenses/by-nc/3.0/</ext-link>
) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
</license>
</permissions>
<abstract>
<p>Squamous cell carcinoma (SCC) arising within the lining of an odontogenic keratocyst (OKC) is a rare occurrence. Although potentially locally destructive, OKC is a benign odontogenic process that typically presents with clinical and radiographic features characteristic of a benign intraosseous neoplasm. We present the clinical and radiographic features of a maxillary mass that demonstrated SCC arising from the lining of an OKC. Although the initial clinical and radiographic presentation suggested an infection or malignant neoplasm, biopsies revealed an infiltrative well-differentiated SCC contiguous with and arising from the focus of a pre-existing OKC. The patient subsequently underwent a type II hemi-maxillectomy with neoadjuvant chemoradiation. This report discusses the clinical and radiographic features associated with intraosseous malignancies, especially those arising from an otherwise benign odontogenic lesion. While the majority of OKCs are benign, the current report illustrates the potential for carcinomatous transformation within the lining of an OKC.</p>
</abstract>
<kwd-group>
<kwd>Carcinoma, Squamous Cell</kwd>
<kwd>Odontogenic Cysts</kwd>
<kwd>Maxilla</kwd>
<kwd>Cell Transformation, Neoplastic</kwd>
</kwd-group>
</article-meta>
</front>
<floats-group>
<fig id="F1" orientation="portrait" position="float">
<label>Fig. 1</label>
<caption>
<title>Preoperative panoramic radiograph demonstrates extensive bone destruction in the right maxilla. The lesion is ill-defined, with ‘blow-out’ of much of the right antrum, also involving the right orbital floor. The maxillary canine is impacted by a pericoronal radiolucency that appears well defined.</title>
</caption>
<graphic xlink:href="isd-47-135-g001"></graphic>
</fig>
<fig id="F2" orientation="portrait" position="float">
<label>Fig. 2</label>
<caption>
<title>A and B. Axial bone window and coronal soft tissue algorithms of CT show the impacted right maxillary canine surrounded by a large, well defined, low intensity lesion growing more along the bone than buccal palatal expansion, extending through the palate and expanding and thinning the buccal cortical border adjacent to the right canine. C and D. Axial bone window and coronal soft tissue algorithm of CT show erosion of much of the floor, anterior and lateral walls of the right maxillary sinus, obliteration of the entire right antral cavity and invasion of the surrounding soft tissue by the lesion. The right lateral nasal wall adjacent to inferior concha and ipsilateral orbital floor are also eroded and edema of the superficial soft tissue on the right side is evident.</title>
</caption>
<graphic xlink:href="isd-47-135-g002"></graphic>
</fig>
<fig id="F3" orientation="portrait" position="float">
<label>Fig. 3</label>
<caption>
<title>Squamous cell carcinoma arising in an odontogenic keratocyst. A. Low-power magnification demonstrates the cyst lining and a satellite cyst within the wall. B. A satellite cyst demonstrates classic odontogenic keratocyst lining epithelium. Note the regimented basal cells and the uniform thickness of the epithelium. C. Odontogenic keratocyst lining demonstrates dysplastic changes. Note the nests of SCC arising from and contiguous with the cyst lining. D. A high-power view shows the infiltrative squamous cell carcinoma.</title>
</caption>
<graphic xlink:href="isd-47-135-g003"></graphic>
</fig>
<fig id="F4" orientation="portrait" position="float">
<label>Fig. 4</label>
<caption>
<title>A postoperative axial computed tomography scan reveals total right maxillectomy with right orbital exoneration and free flap reconstruction. No residual tumor or fluid was detected.</title>
</caption>
<graphic xlink:href="isd-47-135-g004"></graphic>
</fig>
</floats-group>
</pmc>
</record>

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