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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Primary malignant melanoma of oral cavity: A report of three rare cases</title><author><name sortKey="Singh, Hanspal" sort="Singh, Hanspal" uniqKey="Singh H" first="Hanspal" last="Singh">Hanspal Singh</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Kumar, Priya" sort="Kumar, Priya" uniqKey="Kumar P" first="Priya" last="Kumar">Priya Kumar</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Augustine, Jeyaseelan" sort="Augustine, Jeyaseelan" uniqKey="Augustine J" first="Jeyaseelan" last="Augustine">Jeyaseelan Augustine</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Urs, Aadithya B" sort="Urs, Aadithya B" uniqKey="Urs A" first="Aadithya B." last="Urs">Aadithya B. Urs</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Gupta, Sunita" sort="Gupta, Sunita" uniqKey="Gupta S" first="Sunita" last="Gupta">Sunita Gupta</name><affiliation><nlm:aff id="aff2"><italic>Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi, India</italic></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">27041909</idno><idno type="pmc">4792064</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4792064</idno><idno type="RBID">PMC:4792064</idno><idno type="doi">10.4103/0976-237X.177094</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">000521</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000521</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Primary malignant melanoma of oral cavity: A report of three rare cases</title><author><name sortKey="Singh, Hanspal" sort="Singh, Hanspal" uniqKey="Singh H" first="Hanspal" last="Singh">Hanspal Singh</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Kumar, Priya" sort="Kumar, Priya" uniqKey="Kumar P" first="Priya" last="Kumar">Priya Kumar</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Augustine, Jeyaseelan" sort="Augustine, Jeyaseelan" uniqKey="Augustine J" first="Jeyaseelan" last="Augustine">Jeyaseelan Augustine</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Urs, Aadithya B" sort="Urs, Aadithya B" uniqKey="Urs A" first="Aadithya B." last="Urs">Aadithya B. Urs</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Gupta, Sunita" sort="Gupta, Sunita" uniqKey="Gupta S" first="Sunita" last="Gupta">Sunita Gupta</name><affiliation><nlm:aff id="aff2"><italic>Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi, India</italic></nlm:aff></affiliation></author></analytic><series><title level="j">Contemporary Clinical Dentistry</title><idno type="ISSN">0976-237X</idno><idno type="eISSN">0976-2361</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>Oral malignant melanoma (OMM) is a rare tumor of melanocytic origin, accounting for 20–30% of malignant melanomas at the mucosal surface and 16% intra-orally. Hard palate and maxillary gingiva are the most common involved sites. In this case series, we present varying patterns of presentation of three cases of OMM with one case of distant metastasis. All cases in the current series presented at an advanced stage and died within a year of diagnosis. In conclusion, due to the aggressive clinical course and poor prognosis of this deadly lesion, it is of paramount importance to maintain a high index of suspicion for early detection and diagnosis for any pigmented lesion in the oral cavity.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Auluck, A" uniqKey="Auluck A">A Auluck</name></author><author><name sortKey="Zhang, L" uniqKey="Zhang L">L Zhang</name></author><author><name sortKey="Desai, R" uniqKey="Desai R">R Desai</name></author><author><name sortKey="Rosin, Mp" uniqKey="Rosin M">MP Rosin</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hashemi Pour, Ms" uniqKey="Hashemi Pour M">MS Hashemi Pour</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Umeda, M" uniqKey="Umeda M">M Umeda</name></author><author><name sortKey="Komatsubara, H" uniqKey="Komatsubara H">H Komatsubara</name></author><author><name sortKey="Shigeta, T" uniqKey="Shigeta T">T Shigeta</name></author><author><name sortKey="Ojima, Y" uniqKey="Ojima Y">Y Ojima</name></author><author><name sortKey="Minamikawa, T" uniqKey="Minamikawa T">T Minamikawa</name></author><author><name sortKey="Shibuya, Y" uniqKey="Shibuya Y">Y Shibuya</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Tanaka, N" uniqKey="Tanaka N">N Tanaka</name></author><author><name sortKey="Mimura, M" uniqKey="Mimura M">M Mimura</name></author><author><name sortKey="Ogi, K" uniqKey="Ogi K">K Ogi</name></author><author><name sortKey="Amagasa, T" uniqKey="Amagasa T">T Amagasa</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Barker, Bf" uniqKey="Barker B">BF Barker</name></author><author><name sortKey="Carpenter, Wm" uniqKey="Carpenter W">WM Carpenter</name></author><author><name sortKey="Daniels, Te" uniqKey="Daniels T">TE Daniels</name></author><author><name sortKey="Kahn, Ma" uniqKey="Kahn M">MA Kahn</name></author><author><name sortKey="Leider, As" uniqKey="Leider A">AS Leider</name></author><author><name sortKey="Lozada Nur, F" uniqKey="Lozada Nur F">F Lozada-Nur</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rosenberg, Sa" uniqKey="Rosenberg S">SA Rosenberg</name></author><author><name sortKey="Lotze, Mt" uniqKey="Lotze M">MT Lotze</name></author><author><name sortKey="Muul, Lm" uniqKey="Muul L">LM Muul</name></author><author><name sortKey="Leitman, S" uniqKey="Leitman S">S Leitman</name></author><author><name sortKey="Chang, Ae" uniqKey="Chang A">AE Chang</name></author><author><name sortKey="Ettinghausen, Se" uniqKey="Ettinghausen S">SE Ettinghausen</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ebenezer, J" uniqKey="Ebenezer J">J Ebenezer</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Lucas, Rb" uniqKey="Lucas R">RB Lucas</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Marx, Re" uniqKey="Marx R">RE Marx</name></author><author><name sortKey="Stern, D" uniqKey="Stern D">D Stern</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Contemp Clin Dent</journal-id><journal-id journal-id-type="iso-abbrev">Contemp Clin Dent</journal-id><journal-id journal-id-type="publisher-id">CCD</journal-id><journal-title-group><journal-title>Contemporary Clinical Dentistry</journal-title></journal-title-group><issn pub-type="ppub">0976-237X</issn><issn pub-type="epub">0976-2361</issn><publisher><publisher-name>Medknow Publications & Media Pvt Ltd</publisher-name><publisher-loc>India</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">27041909</article-id><article-id pub-id-type="pmc">4792064</article-id><article-id pub-id-type="publisher-id">CCD-7-87</article-id><article-id pub-id-type="doi">10.4103/0976-237X.177094</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Primary malignant melanoma of oral cavity: A report of three rare cases</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Singh</surname><given-names>Hanspal</given-names></name><xref ref-type="aff" rid="aff1"></xref><xref ref-type="corresp" rid="cor1"></xref></contrib><contrib contrib-type="author"><name><surname>Kumar</surname><given-names>Priya</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Augustine</surname><given-names>Jeyaseelan</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Urs</surname><given-names>Aadithya B.</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Gupta</surname><given-names>Sunita</given-names></name><xref ref-type="aff" rid="aff2">1</xref></contrib></contrib-group><aff id="aff1"><italic>Department of Oral and Maxillofacial Pathology, Maulana Azad Institute of Dental Sciences, New Delhi, India</italic></aff><aff id="aff2"><label>1</label><italic>Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi, India</italic></aff><author-notes><corresp id="cor1"><bold>Correspondence:</bold> Dr. Hanspal Singh, Department of Oral and Maxillofacial Pathology, Maulana Azad Institute of Dental Sciences, New Delhi, India. E-mail: <email xlink:href="drhans007@gmail.com">drhans007@gmail.com</email></corresp></author-notes><pub-date pub-type="ppub"><season>Jan-Mar</season><year>2016</year></pub-date><volume>7</volume><issue>1</issue><fpage>87</fpage><lpage>89</lpage><permissions><copyright-statement>Copyright: © Contemporary Clinical Dentistry</copyright-statement><copyright-year>2016</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-sa/3.0"><license-p>This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.</license-p></license></permissions><abstract><p>Oral malignant melanoma (OMM) is a rare tumor of melanocytic origin, accounting for 20–30% of malignant melanomas at the mucosal surface and 16% intra-orally. Hard palate and maxillary gingiva are the most common involved sites. In this case series, we present varying patterns of presentation of three cases of OMM with one case of distant metastasis. All cases in the current series presented at an advanced stage and died within a year of diagnosis. In conclusion, due to the aggressive clinical course and poor prognosis of this deadly lesion, it is of paramount importance to maintain a high index of suspicion for early detection and diagnosis for any pigmented lesion in the oral cavity.</p></abstract><kwd-group><kwd>Malignant melanoma</kwd><kwd>metastasis</kwd><kwd>oral melanoma</kwd></kwd-group></article-meta></front><body><sec sec-type="intro" id="sec1-1"><title>Introduction</title><p>Oral malignant melanoma (OMM) is a malignant neoplasm of melanocytic origin.[<xref rid="ref1" ref-type="bibr">1</xref>] Indian studies have revealed that 20–30% of malignant melanoma are at the mucosal surface and 16% are intraoral.[<xref rid="ref2" ref-type="bibr">2</xref>]</p><p>In this case series, we present varying patterns of presentation of OMM with one case of distant metastasis.</p></sec><sec id="sec1-2"><title>Case Reports</title><sec id="sec2-1"><title>Case 1</title><p>A 65-year-old male patient complained of growth and occasional bleeding on the right side of the palate since 8 months. The patient also had a history of smoking 10–12 bidis/day for 5–7 years and occasionally consumed alcohol.</p><p>Clinical examination revealed gross facial asymmetry, with swelling over the right side of the face and upper lip. Bilateral submandibular lymph nodes were palpable and firm in consistency. A proliferative fungating growth was seen on the right side of the edentulous maxillary arch and palate, extending over the right alveolar ridge and facial vestibule up to the midline anteriorly, and covering the hard and soft palate up to the tuberosity posteriorly [<xref ref-type="fig" rid="F1">Figure 1a</xref>]. The growth was brownish-gray with black and red patches, representing necrotic and hemorrhagic areas. Soft palate showed the presence of 4–5 brownish macules, each 0.5 cm in diameter [<xref ref-type="fig" rid="F1">Figure 1b</xref>].</p><fig id="F1" position="float"><label>Figure 1</label><caption><p>(a) Blackish brown proliferative growth on edentulous maxillary arch and palate and (b) small blackish macules on the soft palate. Contrast enhanced computed tomography showing (c) a large infiltrating homogenous mass on right anterior alveolus. (d) heterogenous enlarged submandibular lymph nodes</p></caption><graphic xlink:href="CCD-7-87-g001"></graphic></fig><p>The contrast enhanced computed tomography (CT) maxilla showed large homogenous mass with infiltrating margins in the right anterior alveolus extending into the hard palate posteriorly [<xref ref-type="fig" rid="F1">Figure 1c</xref>] and skin of the upper lip anteriorly with enlarged right submandibular lymph node [<xref ref-type="fig" rid="F1">Figure 1d</xref>]. Histopathology showed stroma invaded by melanocytes displaying pleomorphism [<xref ref-type="fig" rid="F2">Figure 2a</xref>]. The melanocytes were predominantly spindle-shaped and exhibited mitotic figures. Epitheloid shaped melanocytes were also seen dispersed in between. The growth pattern of these melanocytes was both in the radial and vertical growth phases. Intertwining and fasciculated bundles of malignant melanocytes [<xref ref-type="fig" rid="F2">Figure 2b</xref>] were seen in a streaming fashion.</p><fig id="F2" position="float"><label>Figure 2</label><caption><p>Microphotograph showing Case number 1 (a) pleomorphic melanocytes (H and E, ×100) and (b) intertwining and fasciculated bundles of malignant melanocytes of (H and E, ×400). Case number 2 (c) showing pleomorphic dysplastic melanocytic cells arranged in a pagetoid pattern (H and E, ×100) and (d) epithelioid cells interspersed with the pigmented cells (H and E, ×400). Case number 3 showing (e and f) atypical melanocytes singly and in nests in pagetoid fashion and in sheets (e: H and E, ×40; f: H and E, ×100)</p></caption><graphic xlink:href="CCD-7-87-g002"></graphic></fig></sec><sec id="sec2-2"><title>Case 2</title><p>A 57-year-old male patient presented with swelling on left side of upper jaw which was tender to touch. The patient was apparently well 1 month ago when he noticed pain in upper left upper back tooth region and swelling on left side of face which gradually increased. The patient was chronic beedi smoker with occasional consumption of alcohol since 35–40 years.</p><p>On inspection, extraorally, a diffuse swelling measuring 6 cm × 5 cm in size extending superiorly from 2 cm below the infraorbital margin till the corner of mouth inferiorly was seen. Intraorally, brownish black, irregular growth of 6 cm × 4 cm was seen extending from 13 to 27 on both buccal and lingual aspects involving ridge area [Figure <xref ref-type="fig" rid="F3">3a</xref> and <xref ref-type="fig" rid="F3">b</xref>]. On palpation, extraoral swelling was soft fluctuant and tender. Intraorally, swelling was soft to firm inconsistency and tender.</p><fig id="F3" position="float"><label>Figure 3</label><caption><p>(a and b) Brownish black, irregular growth extending from 13 to 27 on both buccal and lingual aspects involving ridge area. Contrast enhanced computed tomography showing (c) a homogeneous mass with the erosion of left alveolar arch and (d) extension into the left lateral margin of hard palate</p></caption><graphic xlink:href="CCD-7-87-g003"></graphic></fig><p>Radiographically, a homogeneously enhancing soft tissue mass was seen involving left gingivoalveolar complex with the erosion of left alveolar arch [<xref ref-type="fig" rid="F3">Figure 3c</xref>], extension into the left lateral margin of hard palate [<xref ref-type="fig" rid="F3">Figure 3d</xref>] with the destruction of inferolateral wall of left maxillary sinus. Histopathologically, the connective tissue showed numerous ovoid to stellate dysplastic cells showing pleomorphism and dense dark brown pigmented granules throughout the cytoplasm arranged in a pagetoid pattern. Numerous epithelioid cells were seen interspersed with the pigmented cells. The connective tissue was composed of loosely arranged collagen fibers with moderate vascularity. The surface epithelium was ulcerated [Figure <xref ref-type="fig" rid="F2">2c</xref> and <xref ref-type="fig" rid="F2">d</xref>].</p></sec><sec id="sec2-3"><title>Case 3</title><p>A 55-year-old male patient complained of blackish colored growth in the front upper region of the mouth. About 2 months before, he noticed a small black swelling in the anterior edentulous maxillary region of the palate with slight pain. Later, it expanded on the buccal area as well and caused the exfoliation of mobile tooth. The patient was a chronic alcoholic and beedi smoker since 45 years and smoked 20 beedis (approximately) per day. Extra-oral examination revealed the slight fullness of the upper lip. Left submandibular lymph node was enlarged and fixed, nontender, causing asymmetry of face. Intra-oral examination showed grayish black swelling measuring 4 cm × 4 cm × 3 cm in size in the edentulous area with buccal extension of 1.5 cm and palatal extension of 2.5 cm. The growth was well circumscribed ovoid, soft, and nontender [<xref ref-type="fig" rid="F4">Figure 4a</xref>]. CT scan showed heterogenous soft tissue mass perforating the anterior hard palate [<xref ref-type="fig" rid="F4">Figure 4b</xref>]. Borders were irregular and lobulated. A small well-circumscribed round flattened blue-black swelling was observed on the left side of the soft palate. A blackish mass measuring 1 cm was present on the patient's back. Fine needle aspiration cytology, an inky black aspirate was obtained that showed dispersed degenerated large atypical cells with few macronuclei. Histopathological examination showed infiltration of atypical melanocytes singly and in nests, in a pagetoid and organoid fashion [Figure <xref ref-type="fig" rid="F2">2e</xref> and <xref ref-type="fig" rid="F2">f</xref>] showing granular pigmentation and hyperchromatism. The whole-body <sup>18</sup>F-fluorodeoxyglucose positron emission tomography-computed tomography scan showed metastatic deposits in lymph node (cervical, supraclavicular, mediastinal, and abdominal), liver, lung, and brain [Figure <xref ref-type="fig" rid="F4">4c</xref> and <xref ref-type="fig" rid="F4">d</xref>].</p><fig id="F4" position="float"><label>Figure 4</label><caption><p>(a) Well circumscribed ovoid growth on maxillary anterior region. (b) Contrast enhanced computed tomography showing heterogenous soft tissue mass perforating the anterior hard palate. (c and d) <sup>18</sup>F-fluorodeoxyglucose positron emission tomography-computed tomography scan showing metastatic deposits in lymph nodes (cervical, supraclavicular, mediastinal, and abdominal), liver, lung, and brain. Inset shows multiple deposits in the brain</p></caption><graphic xlink:href="CCD-7-87-g004"></graphic></fig></sec></sec><sec sec-type="discussion" id="sec1-3"><title>Discussion</title><p>OMM by nature is asymptomatic and hence their progression may remain unnoticed by patients, contributing to delay in diagnosis.[<xref rid="ref3" ref-type="bibr">3</xref>] All cases in the current series, presented at an advanced stage and were clinically of pigmented mixed type.[<xref rid="ref4" ref-type="bibr">4</xref>] Clinicians should be vigilant toward findings such as swelling within a pigmented area, hemorrhage, interference with denture fitting, and/or loosening of teeth. The absence of indurated edges that are usually indicative of carcinoma may delay diagnosis. Pain is encountered mostly in advanced stages.[<xref rid="ref5" ref-type="bibr">5</xref>] In the present series, two cases presented with pain and one showed hemorrhage as oral manifestation. The third case showed loosening of anterior teeth associated with a pigmented swelling.</p><p>All three patients underwent multimodal chemotherapy and were dead within 6–12 months of diagnosis. Over half of all recurrences/metastasis occur within 3 years. Hence, there is a need to concentrate follow-up in the early time period following diagnosis.[<xref rid="ref6" ref-type="bibr">6</xref>]</p><p>The poor prognosis of OMM with the 5-year survival rate being between 15% and 38%.[<xref rid="ref7" ref-type="bibr">7</xref>] Metastasis from OMM occurs to the regional lymph nodes and in such distant sites as the lung, liver, brain, and bone.[<xref rid="ref8" ref-type="bibr">8</xref>]</p><p>Marx <italic>et al</italic>. recommended chest X-ray after every 6 months, postsurgery as a necessary follow-up tool to assess distant metastasis.[<xref rid="ref9" ref-type="bibr">9</xref>]</p><sec id="sec2-4"><title>Financial support and sponsorship</title><p>Nil.</p></sec><sec id="sec2-5"><title>Conflicts of interest</title><p>There are no conflicts of interest.</p></sec></sec></body><back><ref-list><ref id="ref1"><label>1</label><element-citation publication-type="journal"><person-group 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