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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Inflammatory myofibroblastic tumor of maxilla showing sarcomatous change in an edentulous site with a history of tooth extraction following periodontitis: A case report with discussion</title><author><name sortKey="Biniraj, K R" sort="Biniraj, K R" uniqKey="Biniraj K" first="K. R." last="Biniraj">K. R. Biniraj</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Janardhanan, Mahija" sort="Janardhanan, Mahija" uniqKey="Janardhanan M" first="Mahija" last="Janardhanan">Mahija Janardhanan</name><affiliation><nlm:aff id="aff2"><italic>Department of Oral Pathology and Microbiology, Amrita School of Dentistry, Kochi, Kerala, India</italic></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">25024554</idno><idno type="pmc">4095633</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4095633</idno><idno type="RBID">PMC:4095633</idno><idno type="doi">10.4103/0972-124X.134582</idno><date when="2014">2014</date><idno type="wicri:Area/Pmc/Corpus">000198</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000198</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Inflammatory myofibroblastic tumor of maxilla showing sarcomatous change in an edentulous site with a history of tooth extraction following periodontitis: A case report with discussion</title><author><name sortKey="Biniraj, K R" sort="Biniraj, K R" uniqKey="Biniraj K" first="K. R." last="Biniraj">K. R. Biniraj</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Janardhanan, Mahija" sort="Janardhanan, Mahija" uniqKey="Janardhanan M" first="Mahija" last="Janardhanan">Mahija Janardhanan</name><affiliation><nlm:aff id="aff2"><italic>Department of Oral Pathology and Microbiology, Amrita School of Dentistry, Kochi, Kerala, India</italic></nlm:aff></affiliation></author></analytic><series><title level="j">Journal of Indian Society of Periodontology</title><idno type="ISSN">0972-124X</idno><idno type="eISSN">0975-1580</idno><imprint><date when="2014">2014</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Inflammatory myofibroblastic tumor (IMT) is a rare tumor of uncertain origin with variable biological behavior ranging from reactive lesions to highly aggressive malignancy. Oral IMTs are extremely rare and only 25 cases had been reported so far. A case of IMT with sarcomatous transformation in an extraction site with a history of tooth extraction following tooth mobility of an upper left molar tooth is presented here. The tooth was extracted following a complaint of gingival swelling and mobility of tooth. Though malignant transformation in IMTs had been documented in the extra oral sites, wide search of associated literature suggests, this is the first case of oral IMT showing malignant change associated with gingiva. The case report attempts to highlight the variant possibilities of tooth mobility other than periodontitis and the importance of assessing the primary cause of such conditions.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Coffin, Cm" uniqKey="Coffin C">CM Coffin</name></author><author><name sortKey="Watterson, J" uniqKey="Watterson J">J Watterson</name></author><author><name sortKey="Priest, Jr" uniqKey="Priest J">JR Priest</name></author><author><name sortKey="Dehner, Lp" uniqKey="Dehner L">LP Dehner</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Shek, Aw" uniqKey="Shek A">AW Shek</name></author><author><name sortKey="Wu, Pc" uniqKey="Wu P">PC Wu</name></author><author><name sortKey="Samman, N" uniqKey="Samman N">N Samman</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Binmadi, No" uniqKey="Binmadi N">NO Binmadi</name></author><author><name sortKey="Packman, H" uniqKey="Packman H">H Packman</name></author><author><name sortKey="Papadimitriou, Jc" uniqKey="Papadimitriou J">JC Papadimitriou</name></author><author><name sortKey="Scheper, M" uniqKey="Scheper M">M Scheper</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Biselli, R" uniqKey="Biselli R">R Biselli</name></author><author><name sortKey="Boldrini, R" uniqKey="Boldrini R">R Boldrini</name></author><author><name sortKey="Ferlini, C" uniqKey="Ferlini C">C Ferlini</name></author><author><name sortKey="Boglino, C" uniqKey="Boglino C">C Boglino</name></author><author><name sortKey="Inserra, A" uniqKey="Inserra A">A Inserra</name></author><author><name sortKey="Bosman, C" uniqKey="Bosman C">C Bosman</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Coffin, Cm" uniqKey="Coffin C">CM Coffin</name></author><author><name sortKey="Dehner, Lp" uniqKey="Dehner L">LP Dehner</name></author><author><name sortKey="Meis Kindblom, Jm" uniqKey="Meis Kindblom J">JM Meis-Kindblom</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Poh, Cf" uniqKey="Poh C">CF Poh</name></author><author><name sortKey="Priddy, Rw" uniqKey="Priddy R">RW Priddy</name></author><author><name sortKey="Dahlman, Dm" uniqKey="Dahlman D">DM Dahlman</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Brooks, Jk" uniqKey="Brooks J">JK Brooks</name></author><author><name sortKey="Nikitakis, Ng" uniqKey="Nikitakis N">NG Nikitakis</name></author><author><name sortKey="Frankel, Bf" uniqKey="Frankel B">BF Frankel</name></author><author><name sortKey="Papadimitriou, Jc" uniqKey="Papadimitriou J">JC Papadimitriou</name></author><author><name sortKey="Sauk, Jj" uniqKey="Sauk J">JJ Sauk</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Naveen, J" uniqKey="Naveen J">J Naveen</name></author><author><name sortKey="Sonalika, W" uniqKey="Sonalika W">W Sonalika</name></author><author><name sortKey="Prabhu, S" uniqKey="Prabhu S">S Prabhu</name></author><author><name sortKey="Gopalkrishnan, K" uniqKey="Gopalkrishnan K">K Gopalkrishnan</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Som, Pm" uniqKey="Som P">PM Som</name></author><author><name sortKey="Brandwein, Ms" uniqKey="Brandwein M">MS Brandwein</name></author><author><name sortKey="Maldjian, C" uniqKey="Maldjian C">C Maldjian</name></author><author><name sortKey="Reino, Aj" uniqKey="Reino A">AJ Reino</name></author><author><name sortKey="Lawson, W" uniqKey="Lawson W">W Lawson</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Dishop, Mk" uniqKey="Dishop M">MK Dishop</name></author><author><name sortKey="Warner, Bw" uniqKey="Warner B">BW Warner</name></author><author><name sortKey="Dehner, Lp" uniqKey="Dehner L">LP Dehner</name></author><author><name sortKey="Kriss, Vm" uniqKey="Kriss V">VM Kriss</name></author><author><name sortKey="Greenwood, Mf" uniqKey="Greenwood M">MF Greenwood</name></author><author><name sortKey="Geil, Jd" uniqKey="Geil J">JD Geil</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Zhang, H" uniqKey="Zhang H">H Zhang</name></author><author><name sortKey="Erickson Johnson, M" uniqKey="Erickson Johnson M">M Erickson-Johnson</name></author><author><name sortKey="Wang, X" uniqKey="Wang X">X Wang</name></author><author><name sortKey="Bahrami, A" uniqKey="Bahrami A">A Bahrami</name></author><author><name sortKey="Medeiros, F" uniqKey="Medeiros F">F Medeiros</name></author><author><name sortKey="Lonzo, Ml" uniqKey="Lonzo M">ML Lonzo</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">J Indian Soc Periodontol</journal-id><journal-id journal-id-type="iso-abbrev">J Indian Soc Periodontol</journal-id><journal-id journal-id-type="publisher-id">JISP</journal-id><journal-title-group><journal-title>Journal of Indian Society of Periodontology</journal-title></journal-title-group><issn pub-type="ppub">0972-124X</issn><issn pub-type="epub">0975-1580</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">25024554</article-id><article-id pub-id-type="pmc">4095633</article-id><article-id pub-id-type="publisher-id">JISP-18-375</article-id><article-id pub-id-type="doi">10.4103/0972-124X.134582</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Inflammatory myofibroblastic tumor of maxilla showing sarcomatous change in an edentulous site with a history of tooth extraction following periodontitis: A case report with discussion</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Biniraj</surname><given-names>K. R.</given-names></name><xref ref-type="aff" rid="aff1"></xref><xref ref-type="corresp" rid="cor1"></xref></contrib><contrib contrib-type="author"><name><surname>Janardhanan</surname><given-names>Mahija</given-names></name><xref ref-type="aff" rid="aff2">1</xref></contrib></contrib-group><aff id="aff1"><italic>Department of Periodontology, Royal Dental College, Palakkad, Chalissery, Kerala, India</italic></aff><aff id="aff2"><label>1</label><italic>Department of Oral Pathology and Microbiology, Amrita School of Dentistry, Kochi, Kerala, India</italic></aff><author-notes><corresp id="cor1"><bold>Address for correspondence:</bold> Dr. K. R. Biniraj, Department of Periodontology, Royal Dental College, Palakkad, Chalissery P.O, Kerala - 679 536, India. E-mail: <email xlink:href="binirajkr@gmail.com">binirajkr@gmail.com</email></corresp></author-notes><pub-date pub-type="ppub"><season>May-Jun</season><year>2014</year></pub-date><volume>18</volume><issue>3</issue><fpage>375</fpage><lpage>378</lpage><history><date date-type="received"><day>14</day><month>9</month><year>2013</year></date><date date-type="accepted"><day>25</day><month>11</month><year>2013</year></date></history><permissions><copyright-statement>Copyright: © Journal of Indian Society of Periodontology</copyright-statement><copyright-year>2014</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-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Inflammatory myofibroblastic tumor (IMT) is a rare tumor of uncertain origin with variable biological behavior ranging from reactive lesions to highly aggressive malignancy. Oral IMTs are extremely rare and only 25 cases had been reported so far. A case of IMT with sarcomatous transformation in an extraction site with a history of tooth extraction following tooth mobility of an upper left molar tooth is presented here. The tooth was extracted following a complaint of gingival swelling and mobility of tooth. Though malignant transformation in IMTs had been documented in the extra oral sites, wide search of associated literature suggests, this is the first case of oral IMT showing malignant change associated with gingiva. The case report attempts to highlight the variant possibilities of tooth mobility other than periodontitis and the importance of assessing the primary cause of such conditions.</p></abstract><kwd-group><kwd>Inflammatory myofibroblastic tumor</kwd><kwd>malignant transformation</kwd><kwd>maxillary sinus</kwd><kwd>oral</kwd></kwd-group></article-meta></front><body><sec sec-type="intro" id="sec1-1"><title>INTRODUCTION</title><p>The term inflammatory myofibroblastic tumor (IMT) denotes a wide spectrum of pathological entities ranging from reactive to neoplastic lesions. IMT is most commonly found in the lung, abdomen, retroperitoneum and extremities.</p><p>Though initially considered as a primary pathology of lung, these lesions can be found virtually at any anatomic site, most common extra pulmonary sites of involvement being liver and orbit. Head and neck IMTs are rare and make up only around 14% of the extra pulmonary site.[<xref rid="ref1" ref-type="bibr">1</xref>] Ever since the first report of oral lesion in 1981,[<xref rid="ref2" ref-type="bibr">2</xref>] until date only 25 cases had been published and none of these cases showed any evidence of recurrence, malignant transformation, metastasis or death.[<xref rid="ref3" ref-type="bibr">3</xref>] This is a rare case of IMT showing sarcomatous change in a patient who presented with a swelling in the maxillary alveolar ridge following extraction of a mobile tooth.</p></sec><sec id="sec1-2"><title>CASE REPORT</title><p>The present case report is about a 38-year-old female patient who presented with the complaint of a painless swelling of the upper left posterior alveolar ridge. She gave a history of extraction of a mobile upper left second molar tooth from that area 2 months ago, which had gingival swelling and was progressively mobile. On intra oral examination, a swelling of 3 cm × 4 cm was noticed on the alveolar ridge at the site of extraction in 27 region. The swelling was normal in color with smooth surface except in one area where the indentation from the opposing tooth was noticed [<xref ref-type="fig" rid="F1">Figure 1</xref>]. The swelling started to increase following tooth extraction and started to impinge on lower tooth. Frequent lacrimation from the left eye and difficulty in breathing through the left nostril were present since 3 months. Extra oral examination revealed mild facial asymmetry due to the presence of a diffuse swelling in the left maxillary region. The orbit was pushed upwards and the left lateral ala of nose was raised [<xref ref-type="fig" rid="F2">Figure 2</xref>].</p><fig id="F1" position="float"><label>Figure 1</label><caption><p>Intra oral presentation of the tumor at the site of extraction</p></caption><graphic xlink:href="JISP-18-375-g001"></graphic></fig><fig id="F2" position="float"><label>Figure 2</label><caption><p>Facial asymmetry with raised left orbit</p></caption><graphic xlink:href="JISP-18-375-g002"></graphic></fig><sec id="sec2-1"><title>Radiographic features</title><p>Orthopantamograph showed an ill-defined osteolytic lesion in the left posterior maxilla [<xref ref-type="fig" rid="F3">Figure 3</xref>]. Computed tomography image revealed an irregular soft-tissue lesion in the left maxillary sinus causing expansion and thinning of medial wall and erosion of anterior, inferior and superior walls. Intraorbital extension of the lesion was noticed with the involvement of inferior rectus muscle and there was extension of the tumor into the ethmoid sinus, nasal cavity, oral cavity, pterigopalatine fossa and infra temporal fossa [<xref ref-type="fig" rid="F4">Figure 4</xref>]. Magnetic resonance imaging image showed intra orally the lesion extended into the left alveolus causing erosion and expansion of alveolus [<xref ref-type="fig" rid="F5">Figure 5</xref>]. Based on the clinical presentation and imaging studies, the lesion was provisionally diagnosed as primary intra-alveolar carcinoma.</p><fig id="F3" position="float"><label>Figure 3</label><caption><p>Orthopantamograph showing an ill-defined radiolucent lesion in the left posterior maxilla</p></caption><graphic xlink:href="JISP-18-375-g003"></graphic></fig><fig id="F4" position="float"><label>Figure 4</label><caption><p>Computed tomography scan showing a soft tissue mass in the maxillary sinus with extension of the tumor into the nasal cavity, ethmoid sinus and orbit</p></caption><graphic xlink:href="JISP-18-375-g004"></graphic></fig><fig id="F5" position="float"><label>Figure 5</label><caption><p>Magnetic resonance imaging T1-weighted image with contrast showing a soft-tissue lesion involving the left maxilla, nasal cavity, orbit and ethmoid sinus with extension into the alveolus and infra temporal fossa</p></caption><graphic xlink:href="JISP-18-375-g005"></graphic></fig></sec><sec id="sec2-2"><title>Histopathologic features</title><p>Left side maxillectomy was performed retaining the orbit. The histological sections from the orbital margin showed sheets of atypical spindle cells with indistinct cytoplasm and large vesicular nuclei showing pleomorphism [<xref ref-type="fig" rid="F6">Figure 6</xref>]. The proliferating spindle cells were admixed with numerous inflammatory cells mainly neutrophils, lymphocytes and plasma cells [<xref ref-type="fig" rid="F7">Figure 7</xref>]. On Immunohistochemical staining, the tumor cells were strongly positive for vimentin and smooth muscle actin (SMA), but negative for CD 1a, S100, Desmin, CD 34, activin receptor-like kinase (ALK)-1 and MPO. Nuclear positivity for the proliferative marker Ki 67 was seen in 8-10% of the tumor cells. CD 68 positive cells were seen focally indicting the presence of histiocytes. Based on the clinical presentation, histopathological features and immunohistochemistry the lesion was diagnosed as IMT with focal sarcomatous change. Following resection, patient underwent radiotherapy. At 6 months later the patient developed recurrence of the lesion and finally succumbed to death.</p><fig id="F6" position="float"><label>Figure 6</label><caption><p>The sections from the orbital margin displaying sheets of atypical spindle cells showing large, pleomorphic nuclei with prominent nucleoli (H and E, ×40)</p></caption><graphic xlink:href="JISP-18-375-g006"></graphic></fig><fig id="F7" position="float"><label>Figure 7</label><caption><p>Fascicles of proliferating spindle cells admixed with numerous inflammatory cells</p></caption><graphic xlink:href="JISP-18-375-g007"></graphic></fig></sec></sec><sec id="sec1-3"><title>DISCUSSION (IMT)</title><p>IMT represents a spectrum of myofibroblastic proliferation with diverse pathogenesis and clinical behaviors, varying from reactive to neoplastic processes. The controversies regarding the true nature of the lesion has led to the use of numerous terminologies such as pseudosarcomatous myofibroblastic proliferation, inflammatory sarcoma, plasma-cell granuloma, inflammatory pseudo tumor and inflammatory histiocytic proliferation to be used to denote this tumor.[<xref rid="ref4" ref-type="bibr">4</xref>] Until 1998 IMTs were considered as indolent reactive lesions and were known as inflammatory pseudo tumors. An aberrant or exaggerated response to tissue injury without an established cause had been favored as the pathogenesis of IMT.[<xref rid="ref5" ref-type="bibr">5</xref>] Recently, the concept of IMT being a benign reactive lesion has been challenged owing to the clinical demonstration of recurrences as high as 37%, the presence of regional metastases and cytogenetic evidence of acquired clonal chromosomal abnormality.[<xref rid="ref6" ref-type="bibr">6</xref>] It has also been suggested that reactive and neoplastic myofibroblastic lesions are two separate entities and the term IMT need to be reserved for neoplastic lesions whereas the term inflammatory pseudotumor (IPT) be retained for reactive lesions.[<xref rid="ref7" ref-type="bibr">7</xref>]</p><p>In the Head and neck region, IMTs are more common in the upper respiratory tract (11% of extra pulmonary cases) and the remainder of the head and neck sites (including the orbit, paranasal sinuses, major salivary glands, thyroid and soft-tissues of the face and neck, in descending order of frequency) make up less than 5%.[<xref rid="ref8" ref-type="bibr">8</xref>] Oral IMTs are extremely rare. Binmadi <italic>et al</italic>. reviewed all the 22 published cases of oral IMTs diagnosed as IPT or IMT including the one reported by them.[<xref rid="ref3" ref-type="bibr">3</xref>] They observed that the tumor mainly involved the soft-tissues of the oral cavity with majority of the cases occurring in the buccal mucosa. Following their report, three additional cases of oral IMTs were documented thereby accounting for the total number of 25 published cases.[<xref rid="ref5" ref-type="bibr">5</xref>] Intrabony IMTs are rare and a literature review on intrabony lesions in the head and neck region showed that majority of lesions occurred in the maxillary sinus.[<xref rid="ref8" ref-type="bibr">8</xref><xref rid="ref9" ref-type="bibr">9</xref><xref rid="ref10" ref-type="bibr">10</xref><xref rid="ref11" ref-type="bibr">11</xref>] Out of the 16 documented cases of IMTS involving maxillary sinus three cases showed extension of the lesion into the oral cavity presenting as intraoral tumors.[<xref rid="ref2" ref-type="bibr">2</xref><xref rid="ref8" ref-type="bibr">8</xref><xref rid="ref9" ref-type="bibr">9</xref>]</p><p>The diagnosis of IMTs depends exclusively on the microscopic appearance of the lesional tissue. Histopathological resemblance of inflammatory fibroblastic lesions and spindle cell neoplasms makes the categorization of this tumor difficult and its diagnosis more challenging. The use of myofibroblastic markers seems to be promising but no consistency in the expressivity of these markers were noticed. However, in most cases there was strong diffuse staining of tumor cells with vimentin and less commonly with myogenic markers like SMA, muscle specific actin, ALK-1 and Desmin. CD 68 positivity is noticed in 25% of the cases.[<xref rid="ref1" ref-type="bibr">1</xref>] In this case, the lesional tissue consisted of predominantly bland spindle cells which showed strong vimentin and SMA positivity. The presence of chronic inflammatory infiltrate consisting of neutrophils, lymphocytes, plasma cells and CD68 positive macrophages admixed with tumor cells was a consistent finding. A very unusual finding was the cytological atypia with nuclear pleomorphism and increased mitotic activity consistent with malignant transformation seen in the lesion, which otherwise appeared to be reactive in origin. Positivity for the nuclear protein ki 67 also indicated the aggressive nature of this tumor. Though malignant transformation in IMTs had been observed in other regions no evidence of tumor recurrence or malignant transformation was reported in oral IMTs.[<xref rid="ref10" ref-type="bibr">10</xref><xref rid="ref11" ref-type="bibr">11</xref>]</p></sec><sec id="sec1-4"><title>DISCUSSION (CASE REPORT)</title><p>In the present case, the lesion was noticed at the site of extraction in the maxillary alveolar ridge. Taking into consideration the role played by myofibroblast in the formation of granulation tissue and wound healing, the occurrence of IMTS at extraction site might be taken as a direct evidence for the reactive nature of the tumor. However, it is more likely that mobility in the tooth which was extracted resulted from the destruction of alveolar bone due to the preexisting tumor and the trauma of extraction would have accelerated the growth of the tumor leading to the sudden clinical manifestation.</p><p>In our patient the tumor of maxillary sinus showed extension into the nasal cavity, orbit, ethmoid sinus, infratemporal fossa, pterigopalatine fossa and oral cavity. The clinical symptoms like epiphora, nasal congestion and difficulty in breathing seen in this patient could be attributed to the extension of tumor into the orbit and nasal cavity.</p></sec><sec sec-type="conclusions" id="sec1-5"><title>CONCLUSIONS</title><p>Tooth mobility invariably result from weakening of tooth supporting periodontal tissues, but it need not always be a resultant of diseases of periodontium. This present case is a clear example of rare diseases that involves tooth supporting areas as well as the basal bone that could manifest as mobility of involved tooth. A tooth presenting with grade 3 mobility is generally assumed to be periodontally weak and is considered for extraction. Here, the mobility of tooth and the trauma of extraction are suspected to have contributed to the accelerated growth of this reactive tumor. 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