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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Glandular odontogenic cyst of the maxilla: a case report and literature review</title><author><name sortKey="Figueiredo, Nigel Roque" sort="Figueiredo, Nigel Roque" uniqKey="Figueiredo N" first="Nigel Roque" last="Figueiredo">Nigel Roque Figueiredo</name><affiliation><nlm:aff id="aff0001">Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</nlm:aff></affiliation></author><author><name sortKey="Dinkar, Ajit Dattatray" sort="Dinkar, Ajit Dattatray" uniqKey="Dinkar A" first="Ajit Dattatray" last="Dinkar">Ajit Dattatray Dinkar</name><affiliation><nlm:aff id="aff0001">Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</nlm:aff></affiliation></author><author><name sortKey="Khorate, Manisha Maruti" sort="Khorate, Manisha Maruti" uniqKey="Khorate M" first="Manisha Maruti" last="Khorate">Manisha Maruti Khorate</name><affiliation><nlm:aff id="aff0001">Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">28292079</idno><idno type="pmc">5325494</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5325494</idno><idno type="RBID">PMC:5325494</idno><idno type="doi">10.11604/pamj.2016.25.116.10879</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">001F61</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">001F61</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Glandular odontogenic cyst of the maxilla: a case report and literature review</title><author><name sortKey="Figueiredo, Nigel Roque" sort="Figueiredo, Nigel Roque" uniqKey="Figueiredo N" first="Nigel Roque" last="Figueiredo">Nigel Roque Figueiredo</name><affiliation><nlm:aff id="aff0001">Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</nlm:aff></affiliation></author><author><name sortKey="Dinkar, Ajit Dattatray" sort="Dinkar, Ajit Dattatray" uniqKey="Dinkar A" first="Ajit Dattatray" last="Dinkar">Ajit Dattatray Dinkar</name><affiliation><nlm:aff id="aff0001">Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</nlm:aff></affiliation></author><author><name sortKey="Khorate, Manisha Maruti" sort="Khorate, Manisha Maruti" uniqKey="Khorate M" first="Manisha Maruti" last="Khorate">Manisha Maruti Khorate</name><affiliation><nlm:aff id="aff0001">Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</nlm:aff></affiliation></author></analytic><series><title level="j">The Pan African Medical Journal</title><idno type="eISSN">1937-8688</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Glandular Odontogenic Cyst is a relatively rare cyst of odontogenic origin, which shows glandular or salivary features that are thought to indicate the pluripotentiality of odontogenic epithelium. It is seen in middle-aged adults, and commonly involves the anterior region of the jaws, especially the mandible. It shows non-specific clinico-radiographic findings which may resemble other lesions, but has characteristic histopathologic features which help in its diagnosis. This paper reports an unusual presentation of a glandular odontogenic cyst which was diagnosed in a 64-year old female in the posterior maxilla, along with a literature review of this cyst, especially the cases reported in India in the past.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Osny, Fj" uniqKey="Osny F">FJ Osny</name></author><author><name sortKey="Azavedo, Lr" uniqKey="Azavedo L">LR Azavedo</name></author><author><name sortKey="Sant Na, E" uniqKey="Sant Na E">E Sant’Ana</name></author><author><name sortKey="Lara, Vs" uniqKey="Lara V">VS Lara</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Purohit, S" uniqKey="Purohit S">S Purohit</name></author><author><name sortKey="Shah, V" uniqKey="Shah V">V Shah</name></author><author><name sortKey="Bhakhar, V" uniqKey="Bhakhar V">V Bhakhar</name></author><author><name sortKey="Harsh, A" uniqKey="Harsh A">A 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<front><journal-meta><journal-id journal-id-type="nlm-ta">Pan Afr Med J</journal-id><journal-id journal-id-type="iso-abbrev">Pan Afr Med J</journal-id><journal-id journal-id-type="publisher-id">PAMJ</journal-id><journal-title-group><journal-title>The Pan African Medical Journal</journal-title></journal-title-group><issn pub-type="epub">1937-8688</issn><publisher><publisher-name>The African Field Epidemiology Network</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">28292079</article-id><article-id pub-id-type="pmc">5325494</article-id><article-id pub-id-type="publisher-id">PAMJ-25-116</article-id><article-id pub-id-type="doi">10.11604/pamj.2016.25.116.10879</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Glandular odontogenic cyst of the maxilla: a case report and literature review</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Figueiredo</surname><given-names>Nigel Roque</given-names></name><xref ref-type="aff" rid="aff0001">1</xref><xref ref-type="corresp" rid="cor1">&</xref></contrib><contrib contrib-type="author"><name><surname>Dinkar</surname><given-names>Ajit Dattatray</given-names></name><xref ref-type="aff" rid="aff0001">1</xref></contrib><contrib contrib-type="author"><name><surname>Khorate</surname><given-names>Manisha Maruti</given-names></name><xref ref-type="aff" rid="aff0001">1</xref></contrib></contrib-group><aff id="aff0001"><label>1</label>Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</aff><author-notes><corresp id="cor1"><label>&</label>Corresponding author: Nigel Roque Figueiredo, Oral Medicine, Diagnosis and Radiology Department, Goa Dental College & Hospital, Bambolim-Goa, India</corresp></author-notes><pub-date pub-type="epub"><day>26</day><month>10</month><year>2016</year></pub-date><pub-date pub-type="collection"><year>2016</year></pub-date><volume>25</volume><elocation-id>116</elocation-id><history><date date-type="received"><day>05</day><month>10</month><year>2016</year></date><date date-type="accepted"><day>17</day><month>10</month><year>2016</year></date></history><permissions><copyright-statement>© Nigel Roque Figueiredo et al.</copyright-statement><copyright-year>2016</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/2.0/"><license-p>The Pan African Medical Journal - ISSN 1937-8688. This is an Open Access article distributed under the terms of the Creative Commons Attribution License which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Glandular Odontogenic Cyst is a relatively rare cyst of odontogenic origin, which shows glandular or salivary features that are thought to indicate the pluripotentiality of odontogenic epithelium. It is seen in middle-aged adults, and commonly involves the anterior region of the jaws, especially the mandible. It shows non-specific clinico-radiographic findings which may resemble other lesions, but has characteristic histopathologic features which help in its diagnosis. This paper reports an unusual presentation of a glandular odontogenic cyst which was diagnosed in a 64-year old female in the posterior maxilla, along with a literature review of this cyst, especially the cases reported in India in the past.</p></abstract><kwd-group><kwd>Jaw</kwd><kwd>maxilla</kwd><kwd>odontogenic cyst</kwd></kwd-group></article-meta></front><body><sec sec-type="intro" id="sec1"><title>Introduction</title><p>The Glandular Odontogenic Cyst (GOC) is a rare developmental cyst of the jaws. It was initially described by Padayachee and van Wyk in <bold>1</bold>987, who called it ‘Sialo-Odontogenic Cyst’ due to the presence of mucous cells and pools of mucin in the epithelial lining, and the fact that mucous pools are often lined by eosinophilic cuboidal cells which resemble salivary gland ducts [<xref rid="cit0001" ref-type="bibr">1</xref>]. Gardner et al in 1988 suggested the name ‘Glandular Odontogenic Cyst’ because the cyst wall epithelium was odontogenic and contained mucin elements with absence of salivary tissue [<xref rid="cit0002" ref-type="bibr">2</xref>]. In 1992, GOC was included in the WHO typing of tumors under the term GOC or sialo-odontogenic cyst [<xref rid="cit0003" ref-type="bibr">3</xref>]. An important feature of this cyst is that its recognition on the basis of clinical and radiographic features is practically impossible, and only histopathological examination allows for a certain diagnosis [<xref rid="cit0004" ref-type="bibr">4</xref>]. This case report describes a case of glandular odontogenic cyst in a 64-year old female which was diagnosed and treated in our institution.</p></sec><sec id="sec2"><title>Patient and observation</title><p>A 64-year-old female patient reported to our out-patient department with a chief complaint of swelling over the left side of the face since two months. History revealed that the swelling was initially small in size and had gradually increased to its present size. On extra-oral examination, a diffuse swelling was seen over the left cheek region, extending from the left nasolabial fold area, posteriorly to the malar region and supero-inferiorly from 2 cms below the infra-orbital margin to 1 cm away from the left angle of mouth, measuring around 3 x 2.5 cms. The overlying skin was normal with no evidence of any discharge. On palpation, the swelling was firm in consistency, tender, non-pulsatile and non-compressible, with no local rise in temperature. Intra-oral examination revealed a diffuse swelling in the left maxilla and buccal vestibule, extending from 23 till the 26 region (<xref ref-type="fig" rid="f0001">Figure 1</xref>). The swelling was firm in consistency with a smooth surface and tender on palpation. On examination of the teeth, 23 were restored, while 24 and 25 were missing. No tenderness or mobility of the teeth in the area was noted. An intra-oral periapical radiograph showed a large unilocular radiolucency with a well-defined thin corticated border extending from the distal aspect of 23, upto the 26 region. A maxillary true occlusal view showed a unilocular radiolucent lesion in the 23, 24, 25, 26 region causing expansion of the buccal cortical plate in the affected area (<xref ref-type="fig" rid="f0002">Figure 2</xref>). Panoramic radiography showed a well-defined oval-shaped unilocular radiolucency with thin corticated borders extending from the mesial aspect of 23 up to the 26 region, superiorly involving the maxillary sinus and inferiorly the maxillary alveolar crest. The internal structure was completely radiolucent. There was no evidence of root resorption or displacement of 23 or 26 (<xref ref-type="fig" rid="f0003">Figure 3</xref>).</p><fig id="f0001" orientation="portrait" position="float"><label>Figure 1</label><caption><p>Pre-operative intra-oral view</p></caption><graphic xlink:href="PAMJ-25-116-g001"></graphic></fig><fig id="f0002" orientation="portrait" position="float"><label>Figure 2</label><caption><p>Maxillary true occlusal radiograph showing expansion of buccal cortical plate in region of 23, 24, 25 and 26</p></caption><graphic xlink:href="PAMJ-25-116-g002"></graphic></fig><fig id="f0003" orientation="portrait" position="float"><label>Figure 3</label><caption><p>Panoramic radiograph showing an oval-shaped unilocular radiolucency with thin corticated borders extending from 23 to 26 regions</p></caption><graphic xlink:href="PAMJ-25-116-g003"></graphic></fig><p>Based on the history, clinical and radiographic findings, a provisional diagnosis of a residual cyst was made, with a differential diagnosis of an odontogenic cyst or tumor. The lesion was treated conservatively with careful enucleation and curettage. Histopathological examination showed a cystic lining with a pseudoglandular pattern in some areas, with other areas showing a pseudostratified columnar ciliated epithelium with goblet cells, some mucous pools / crypts (<xref ref-type="fig" rid="f0004">Figure 4</xref>). The connective tissue showed presence of collagen fibers, fibroblasts, blood vessels, areas of osteoid formation, and a mild chronic inflammatory cell infiltrate. Thus, based on the histopathology of the enucleated tissue, a final diagnosis of a glandular odontogenic cyst was made. Post-operative healing was uneventful. The patient has been under regular follow-up for the last 2 years, during which time no recurrence has been noted (<xref ref-type="fig" rid="f0005">Figure 5</xref>, <xref ref-type="fig" rid="f0006">Figure 6</xref>).</p><fig id="f0004" orientation="portrait" position="float"><label>Figure 4</label><caption><p>Photomicrograph showing a cystic lesion with papillary projections lined by pseudostratified columnar epithelium with some mucous pools and pseudoglandular areas (a) H&E stain at 10x magnification, (b) H&E stain at 40x magnification</p></caption><graphic xlink:href="PAMJ-25-116-g004"></graphic></fig><fig id="f0005" orientation="portrait" position="float"><label>Figure 5</label><caption><p>Post-operative intra-oral view</p></caption><graphic xlink:href="PAMJ-25-116-g005"></graphic></fig><fig id="f0006" orientation="portrait" position="float"><label>Figure 6</label><caption><p>Post-operative panoramic radiograph</p></caption><graphic xlink:href="PAMJ-25-116-g006"></graphic></fig></sec><sec sec-type="discussion" id="sec3"><title>Discussion</title><p>GOC is a rare lesion with a frequency rate of only 0.012% to 1.3% of all the jaw cysts [<xref rid="cit0005" ref-type="bibr">5</xref>]. Till date, less than 200 cases of this lesion have been reported in literature. A literature review performed using the search terms “glandular odontogenic cyst”, “sialo-odontogenic cyst” on the PubMed interface yielded only 19 cases of histopathologically confirmed GOC that were reported in India, which have been detailed in <xref ref-type="table" rid="t0001">Table 1</xref>, <xref ref-type="table" rid="t0002">Table 2</xref>. GOC usually occurs in middle-aged adults, most often after the fourth decade of life, with a slight male predilection. It is four times more common in the mandible as compared to the maxilla and has a predilection for the anterior region of the jaws [<xref rid="cit0020" ref-type="bibr">20</xref>]. The present case differs from the literature in that it was diagnosed in a 64-year old female, and occurred in the maxilla extending antero-posteriorly from the canine to the first molar region, which is not commonly seen. Clinically features of this cyst are not specific, with lesions usually presenting as asymptomatic swellings. The radiographic appearance also varies and is not pathognomonic. It may present as a unilocular or multilocular radiolucency, which usually shows well-defined borders [<xref rid="cit0020" ref-type="bibr">20</xref>]. Occasionally, scalloping of the border may be noted, while root resorption and displacement are not commonly seen [<xref rid="cit0003" ref-type="bibr">3</xref>]. Radiographically, GOC may resemble lesions like radicular cyst, keratocystic odontogenic tumor, ameloblastoma and central giant cell granuloma. The radiographic features of our case were similar to those described previously, but were suggestive of a residual cyst primarily due to the absence of teeth 24 and 25. An interesting observation in our literature review of the previous cases of GOC reported in India showed that 7 out of 19 cases were also located in edentulous or partially edentulous areas (where teeth were extracted previously). This could indicate that GOC may possibly grow within the jaws without any symptoms in the initial stages, and cause swelling and cortical expansion only at a later date.</p><table-wrap id="t0001" orientation="portrait" position="float"><label>Table 1</label><caption><p>Clinical data of 19 cases of GOC previously reported in India along with the present case</p></caption><table frame="border" rules="all"><thead><tr><th align="left" rowspan="1" colspan="1">Authors of the Case Report</th><th align="center" rowspan="1" colspan="1">Age of Patient (years)</th><th align="center" rowspan="1" colspan="1">Sex</th><th align="center" rowspan="1" colspan="1">Swelling</th><th align="center" rowspan="1" colspan="1">Pain</th><th align="center" rowspan="1" colspan="1">Location</th></tr></thead><tbody><tr><td align="left" rowspan="1" colspan="1">Baliga et al (1997) [<xref rid="cit0006" ref-type="bibr">6</xref>]</td><td align="center" rowspan="1" colspan="1">26</td><td align="left" rowspan="1" colspan="1">Male</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Left anterior maxilla 21-22</td></tr><tr><td align="left" rowspan="1" colspan="1">Jose et al (2000) [<xref rid="cit0007" ref-type="bibr">7</xref>]</td><td align="center" rowspan="1" colspan="1">18</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Right maxilla 16-17 and maxillary sinus</td></tr><tr><td align="left" rowspan="2" valign="top" colspan="1">Krishnamurthy et al (2009) [<xref rid="cit0005" ref-type="bibr">5</xref>]</td><td align="center" rowspan="1" colspan="1">42</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Anterior mandible, extending from right to left body of mandible 36-45</td></tr><tr><td align="center" rowspan="1" colspan="1">21</td><td align="left" rowspan="1" colspan="1">Male</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Left mandible, from midline till ascending ramus</td></tr><tr><td align="left" rowspan="1" colspan="1">Prabhu et al (2010) [<xref rid="cit0008" ref-type="bibr">8</xref>]</td><td align="center" rowspan="1" colspan="1">47</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Right maxilla 14-18 (missing 16)</td></tr><tr><td align="left" rowspan="1" colspan="1">Rao et al (2010) [<xref rid="cit0009" ref-type="bibr">9</xref>]</td><td align="center" rowspan="1" colspan="1">60</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Left maxilla 26-28 and maxillary sinus (missing 27, 28)</td></tr><tr><td align="left" rowspan="1" colspan="1">Guruprasad et al (2011) [<xref rid="cit0010" ref-type="bibr">10</xref>]</td><td align="center" rowspan="1" colspan="1">17</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Right posterior maxilla and maxillary sinus</td></tr><tr><td align="left" rowspan="1" colspan="1">Amberkar et al (2011) [<xref rid="cit0011" ref-type="bibr">11</xref>]</td><td align="center" rowspan="1" colspan="1">29</td><td align="left" rowspan="1" colspan="1">Male</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Bilateral - right maxilla 14-16 and left maxilla 24-26</td></tr><tr><td align="left" rowspan="1" colspan="1">Purohit et al (2014) [<xref rid="cit0002" ref-type="bibr">2</xref>]</td><td align="center" rowspan="1" colspan="1">30</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Right maxilla 15-17</td></tr><tr><td align="left" rowspan="1" colspan="1">Shah M et al (2014) [<xref rid="cit0012" ref-type="bibr">12</xref>]</td><td align="center" rowspan="1" colspan="1">40</td><td align="left" rowspan="1" colspan="1">Male</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Anterior mandible – left to right side 36-43</td></tr><tr><td align="left" rowspan="1" colspan="1">Tambawala et al (2014) [<xref rid="cit0013" ref-type="bibr">13</xref>]</td><td align="center" rowspan="1" colspan="1">17</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Right mandible – angle and ramus</td></tr><tr><td align="left" rowspan="1" colspan="1">Mittal et al (2015) [<xref rid="cit0014" ref-type="bibr">14</xref>]</td><td align="center" rowspan="1" colspan="1">17</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Left anterior to right posterior mandible 34-47</td></tr><tr><td align="left" rowspan="1" colspan="1">Faisal et al (2015) [<xref rid="cit0015" ref-type="bibr">15</xref>]</td><td align="center" rowspan="1" colspan="1">11</td><td align="left" rowspan="1" colspan="1">Male</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Anterior mandible, extending from right to left body of mandible 36-45</td></tr><tr><td align="left" rowspan="1" colspan="1">Raju et al (2015) [<xref rid="cit0016" ref-type="bibr">16</xref>]</td><td align="center" rowspan="1" colspan="1">65</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Anterior mandible, extending from right to left body of mandible (missing 31, 32, 41, 42)</td></tr><tr><td align="left" rowspan="1" colspan="1">Anchlia et al (2015) [<xref rid="cit0017" ref-type="bibr">17</xref>]</td><td align="center" rowspan="1" colspan="1">63</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Right anterior mandible, 31-45</td></tr><tr><td align="left" rowspan="2" valign="top" colspan="1">Shah A et al (2016) [<xref rid="cit0003" ref-type="bibr">3</xref>]</td><td align="center" rowspan="1" colspan="1">25</td><td align="left" rowspan="1" colspan="1">Male</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Left mandible 35-37 (missing 36)</td></tr><tr><td align="center" rowspan="1" colspan="1">30</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Right mandible 46 (missing 46)</td></tr><tr><td align="left" rowspan="1" colspan="1">Surej Kumar et al (2016) [<xref rid="cit0018" ref-type="bibr">18</xref>]</td><td align="center" rowspan="1" colspan="1">46</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Anterior maxilla 12-13</td></tr><tr><td align="left" rowspan="1" colspan="1">Chandra et al (2016) [<xref rid="cit0019" ref-type="bibr">19</xref>]</td><td align="center" rowspan="1" colspan="1">70</td><td align="left" rowspan="1" colspan="1">Female</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Anterior maxilla 21-23</td></tr><tr><td align="left" rowspan="1" colspan="1"><bold>Present Case</bold></td><td align="center" rowspan="1" colspan="1"><bold>64</bold></td><td align="left" rowspan="1" colspan="1"><bold>Female</bold></td><td align="left" rowspan="1" colspan="1"><bold>Present</bold></td><td align="left" rowspan="1" colspan="1"><bold>Present</bold></td><td align="left" rowspan="1" colspan="1"><bold>Left maxilla 23-26</bold></td></tr></tbody></table></table-wrap><table-wrap id="t0002" orientation="portrait" position="float"><label>Table 2</label><caption><p>Radiographic data of 19 cases of GOC previously reported in India along with the present case</p></caption><table frame="border" rules="all"><thead><tr><th align="left" rowspan="1" colspan="1">Authors of the Case Report</th><th align="center" rowspan="1" colspan="1">Radiographic Appearance</th><th align="center" rowspan="1" colspan="1">Margins / Borders</th><th align="center" rowspan="1" colspan="1">Internal Structure</th><th align="center" rowspan="1" colspan="1">Root Resorption</th><th align="center" rowspan="1" colspan="1">Tooth / Root Displacement</th><th align="center" rowspan="1" colspan="1">Cortical Expansion</th><th align="center" rowspan="1" colspan="1">Impacted Tooth</th></tr></thead><tbody><tr><td align="left" rowspan="1" colspan="1">Baliga et al (1997) [<xref rid="cit0006" ref-type="bibr">6</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, Corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Present 22</td><td align="left" rowspan="1" colspan="1">Palatal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Jose et al (2000) [<xref rid="cit0007" ref-type="bibr">7</xref>]</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Buccal, palatal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="2" valign="top" colspan="1">Krishnamurthy et al (2009) [<xref rid="cit0005" ref-type="bibr">5</xref>]</td><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Edentulous</td><td align="left" rowspan="1" colspan="1">Edentulous</td><td align="left" rowspan="1" colspan="1">Buccal, lingual</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Buccal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Prabhu et al (2010) [<xref rid="cit0008" ref-type="bibr">8</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Buccal, palatal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Rao et al (2010) [<xref rid="cit0009" ref-type="bibr">9</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Buccal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Guruprasad et al (2011) [<xref rid="cit0010" ref-type="bibr">10</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Present 18</td></tr><tr><td align="left" rowspan="1" colspan="1">Amberkar et al (2011) [<xref rid="cit0011" ref-type="bibr">11</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Present 14, 15</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Purohit et al (2014) [<xref rid="cit0002" ref-type="bibr">2</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Present 16</td><td align="left" rowspan="1" colspan="1">Present 15, 17</td><td align="left" rowspan="1" colspan="1">Buccal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Shah M et al (2014) [<xref rid="cit0012" ref-type="bibr">12</xref>]</td><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, Corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Buccal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Tambawala et al (2014) [<xref rid="cit0013" ref-type="bibr">13</xref>]</td><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Present 47</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Buccal, lingual with perforation</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Mittal et al (2015) [<xref rid="cit0014" ref-type="bibr">14</xref>]</td><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Faisal et al (2015) [<xref rid="cit0015" ref-type="bibr">15</xref>]</td><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, Corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Buccal, lingual, with perforation</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Raju et al (2015) [<xref rid="cit0016" ref-type="bibr">16</xref>]</td><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Well-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Anchlia et al (2015) [<xref rid="cit0017" ref-type="bibr">17</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, Corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Present 43</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="2" valign="top" colspan="1">Shah A et al (2016) [<xref rid="cit0003" ref-type="bibr">3</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, Corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Buccal, lingual</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, Corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">--</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">NR</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Surej Kumar et al (2016) [<xref rid="cit0018" ref-type="bibr">18</xref>]</td><td align="left" rowspan="1" colspan="1">Unilocular</td><td align="left" rowspan="1" colspan="1">Well-defined, Corticated</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Absent</td><td align="left" rowspan="1" colspan="1">Present</td><td align="left" rowspan="1" colspan="1">Buccal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1">Chandra et al (2016) [<xref rid="cit0019" ref-type="bibr">19</xref>]</td><td align="left" rowspan="1" colspan="1">Multilocular</td><td align="left" rowspan="1" colspan="1">Ill-defined</td><td align="left" rowspan="1" colspan="1">Radiolucent</td><td align="left" rowspan="1" colspan="1">Edentulous</td><td align="left" rowspan="1" colspan="1">Edentulous</td><td align="left" rowspan="1" colspan="1">Buccal, palatal</td><td align="left" rowspan="1" colspan="1">Absent</td></tr><tr><td align="left" rowspan="1" colspan="1"><bold>Present Case</bold></td><td align="left" rowspan="1" colspan="1"><bold>Unilocular</bold></td><td align="left" rowspan="1" colspan="1"><bold>Well-defined, Corticated</bold></td><td align="left" rowspan="1" colspan="1"><bold>Radiolucent</bold></td><td align="left" rowspan="1" colspan="1"><bold>Absent</bold></td><td align="left" rowspan="1" colspan="1"><bold>Absent</bold></td><td align="left" rowspan="1" colspan="1"><bold>Buccal</bold></td><td align="left" rowspan="1" colspan="1"><bold>Absent</bold></td></tr></tbody></table><table-wrap-foot><fn><p>NR: Not reported</p></fn></table-wrap-foot></table-wrap><p>Histologically, GOC shows certain characteristic features that were subdivided by Kaplan et al into major and minor criteria based on the frequency of occurrence of each feature in the previously reported cases [<xref rid="cit0002" ref-type="bibr">2</xref>]. At least focal presence of the major criteria is mandatory, while the presence of minor criteria further supports the correct diagnosis [<xref rid="cit0003" ref-type="bibr">3</xref>]. The major criteria include a lining of squamous epithelium of varying thickness which may show epithelial ‘spheres’ or ‘whorls’ with absence of basal palisading, cuboid-shaped eosinophilic (‘hobnail’) cells, and intraepithelial mucous / goblet cells or pools, with presence of glandular or duct-like structures [<xref rid="cit0003" ref-type="bibr">3</xref>, <xref rid="cit0013" ref-type="bibr">13</xref>]. Papillary proliferation of the epithelial lining, multicystic appearance, and presence of ciliated or clear cells in the epithelium were included as minor criteria [<xref rid="cit0013" ref-type="bibr">13</xref>]. The present case showed most of the characteristic histopathological features of GOC described above. The histopathological features of GOC have been found to resemble a number of lesions having a wide clinico-pathologic spectrum ranging from other odontogenic cysts like lateral periodontal cyst to malignant neoplasms such as central low-grade mucoepidermoid carcinoma, and hence careful and detailed microscopic examination is essential in arriving at a correct diagnosis [<xref rid="cit0005" ref-type="bibr">5</xref>]. The exact treatment of choice of this lesion remains controversial, and varies from enucleation and curettage to local block excision depending on the size and aggressiveness [<xref rid="cit0001" ref-type="bibr">1</xref>]. Enucleation is preferred for small unilocular lesions, whereas management of large lesions includes enucleation with peripheral ostectomy for unilocular cases and marginal resection or partial jaw resection in multilocular cases [<xref rid="cit0016" ref-type="bibr">16</xref>]. Some of the previous cases reported in literature have been found to have an aggressive potential and hence a long-term follow-up is important to monitor for any signs of recurrence. The present case resembled a residual cyst and was thus treated by enucleation and curettage, and has been kept under regular observation till date, with no evidence of recurrence.</p></sec><sec sec-type="conclusion" id="sec4"><title>Conclusion</title><p>GOC is an uncommon cystic lesion, with less than 200 cases reported world-wide till date. This cyst shows non-specific clinico-radiographic characteristics which may resemble a wide-spectrum of lesions. A final diagnosis can only be obtained after a careful histopathologic examination and a long-term follow-up is required to rule out recurrences.</p></sec></body><back><ack><title>Acknowledgement</title><p>Dr. Anita Spadigam, Professor & Head, and Dr. Anita Dhupar, Professor, Department of Oral and Maxillofacial Pathology, Goa Dental College & Hospital, Bambolim, Goa – India, for providing the histopathological analysis, report and photographs.</p></ack><sec sec-type="COI-statement"><title>Competing interests</title><p>The authors declare no competing interests</p></sec><sec><title>Authors’ contributions</title><p>All authors have read and agreed to the final version of this manuscript and have equally contributed to its content and to the management of the case.</p></sec><ref-list><title>References</title><ref id="cit0001"><label>1</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Osny</surname><given-names>FJ</given-names></name><name><surname>Azavedo</surname><given-names>LR</given-names></name><name><surname>Sant’Ana</surname><given-names>E</given-names></name><name><surname>Lara</surname><given-names>VS</given-names></name></person-group><article-title>Glandular odontogenic cyst: case report and review of the literature</article-title><source>Quintessence Int.</source><year>2004</year><month>5</month><volume>35</volume><issue>5</issue><fpage>385</fpage><lpage>9</lpage><pub-id pub-id-type="pmid">15130079</pub-id></element-citation></ref><ref id="cit0002"><label>2</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Purohit</surname><given-names>S</given-names></name><name><surname>Shah</surname><given-names>V</given-names></name><name><surname>Bhakhar</surname><given-names>V</given-names></name><name><surname>Harsh</surname><given-names>A</given-names></name></person-group><article-title>Glandular odontogenic cyst in maxilla: A case report and literature review</article-title><source>J Oral Maxillofac Pathol.</source><year>2014</year><month>5</month><volume>18</volume><issue>2</issue><fpage>320</fpage><lpage>3</lpage><pub-id pub-id-type="doi">10.4103/0973-029X.140923</pub-id><pub-id pub-id-type="pmid">25328322</pub-id></element-citation></ref><ref id="cit0003"><label>3</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Shah</surname><given-names>AA</given-names></name><name><surname>Sangle</surname><given-names>A</given-names></name><name><surname>Bussari</surname><given-names>S</given-names></name><name><surname>Koshy</surname><given-names>AV</given-names></name></person-group><article-title>Glandular odontogenic cyst: A diagnostic dilemma</article-title><source>Indian J Dent.</source><year>2016</year><season>Jan-Mar</season><volume>7</volume><issue>1</issue><fpage>38</fpage><lpage>4</lpage><pub-id pub-id-type="doi">10.4103/0975-962X.179371</pub-id><pub-id pub-id-type="pmid">27134453</pub-id></element-citation></ref><ref id="cit0004"><label>4</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Luczak</surname><given-names>K</given-names></name><name><surname>Nowak</surname><given-names>R</given-names></name><name><surname>Rzeszutko</surname><given-names>M</given-names></name></person-group><article-title>Glandular Odontogenic Cyst of the Mandible Associated with Impacted Tooth - 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