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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Recurrent unicystic ameloblastoma in a child</title><author><name sortKey="Ponniah, Irulandy" sort="Ponniah, Irulandy" uniqKey="Ponniah I" first="Irulandy" last="Ponniah">Irulandy Ponniah</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">22529589</idno><idno type="pmc">3329690</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3329690</idno><idno type="RBID">PMC:3329690</idno><idno type="doi">10.4103/0973-029X.84513</idno><date when="2011">2011</date><idno type="wicri:Area/Pmc/Corpus">001E86</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">001E86</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Recurrent unicystic ameloblastoma in a child</title><author><name sortKey="Ponniah, Irulandy" sort="Ponniah, Irulandy" uniqKey="Ponniah I" first="Irulandy" last="Ponniah">Irulandy Ponniah</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author></analytic><series><title level="j">Journal of Oral and Maxillofacial Pathology : JOMFP</title><idno type="ISSN">0973-029X</idno><idno type="eISSN">1998-393X</idno><imprint><date when="2011">2011</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Unicystic ameloblastoma (UCA) is a clinical subtype of ameloblastoma that is considered prognostically different. The purpose of this report is to present a case of UCA showing dual radiographic pattern in a child. A detailed study of the lesion was carried out in an 8 year old female child who presented to our department of oral and maxillofacial pathology. Clinical, radiological and histopathological findings were recorded. In March 2005, a painless swelling in the left side of the mandible was noted, which on radiographic examination showed a unilocular radioluceny enclosing the crown of mandibular left permanent second molar, extending between the left first permanent molar and anterior margin of the ramus. Histopathologic diagnosis was UCA. The lesion was treated by enucleation. The patient returned with recurrence in 2009, at this time the lesion radiographically presented as a multilocular radiolucency with a soap bubble appearance, extending between the anterior border of the ramus and second premolar. Histopathologic diagnosis was UCA. The lesion was treated by segmental resection with immediate reconstruction. Although a number of treatment modalities are available to treat UCA, many factors need to be taken into consideration in the treatment of UCA in children.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Robinson, L" uniqKey="Robinson L">L Robinson</name></author><author><name sortKey="Martinez, Mg" uniqKey="Martinez M">MG Martinez</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Philipsen, Hp" uniqKey="Philipsen H">HP Philipsen</name></author><author><name sortKey="Reichart, Pa" uniqKey="Reichart P">PA Reichart</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Eversole, Lr" uniqKey="Eversole L">LR Eversole</name></author><author><name sortKey="Leider, As" uniqKey="Leider A">AS Leider</name></author><author><name sortKey="Strub, D" uniqKey="Strub D">D Strub</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kahn, Ma" uniqKey="Kahn M">MA Kahn</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Macdonald Jankowski, Ds" uniqKey="Macdonald Jankowski D">DS MacDonald-Jankowski</name></author><author><name sortKey="Yeung, R" uniqKey="Yeung R">R Yeung</name></author><author><name sortKey="Lee, Km" uniqKey="Lee K">KM Lee</name></author><author><name sortKey="Li, Tk" uniqKey="Li T">TK Li</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Luo, Hy" uniqKey="Luo H">HY Luo</name></author><author><name sortKey="Li, Tj" uniqKey="Li T">TJ Li</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Oliveira Neto, Hh" uniqKey="Oliveira Neto H">HH Oliveira-Neto</name></author><author><name sortKey="Spindula Filho, Jv" uniqKey="Spindula Filho J">JV Spindula-Filho</name></author><author><name sortKey="Dallara, Mc" uniqKey="Dallara M">MC Dallara</name></author><author><name sortKey="Silva, Cm" uniqKey="Silva C">CM Silva</name></author><author><name sortKey="Mendonca, Ef" uniqKey="Mendonca E">EF Mendonca</name></author><author><name sortKey="Batista, Ac" uniqKey="Batista A">AC Batista</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ackermann, Gl" uniqKey="Ackermann G">GL Ackermann</name></author><author><name sortKey="Altini, M" uniqKey="Altini M">M Altini</name></author><author><name sortKey="Shear, M" uniqKey="Shear M">M Shear</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rosenstein, T" uniqKey="Rosenstein T">T Rosenstein</name></author><author><name sortKey="Pogrel, Ma" uniqKey="Pogrel M">MA Pogrel</name></author><author><name sortKey="Smith, Ra" uniqKey="Smith R">RA Smith</name></author><author><name sortKey="Regezi, Ja" uniqKey="Regezi J">JA Regezi</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Lau, Sl" uniqKey="Lau S">SL Lau</name></author><author><name sortKey="Samman, N" uniqKey="Samman N">N Samman</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Zhang, J" uniqKey="Zhang J">J Zhang</name></author><author><name sortKey="Gu, Z" uniqKey="Gu Z">Z Gu</name></author><author><name sortKey="Jiang, L" uniqKey="Jiang L">L Jiang</name></author><author><name sortKey="Zhao, J" uniqKey="Zhao J">J Zhao</name></author><author><name sortKey="Tian, M" uniqKey="Tian M">M Tian</name></author><author><name sortKey="Zhou, J" uniqKey="Zhou J">J Zhou</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Eckardt, Am" uniqKey="Eckardt A">AM Eckardt</name></author><author><name sortKey="Kokemuller, H" uniqKey="Kokemuller H">H Kokemuller</name></author><author><name sortKey="Flemming, P" uniqKey="Flemming P">P Flemming</name></author><author><name sortKey="Schultze, A" uniqKey="Schultze A">A Schultze</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Pogrel, Ma" uniqKey="Pogrel M">MA Pogrel</name></author><author><name sortKey="Montes, Dm" uniqKey="Montes D">DM Montes</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 Oral Maxillofac Pathol</journal-id><journal-id journal-id-type="iso-abbrev">J Oral Maxillofac Pathol</journal-id><journal-id journal-id-type="publisher-id">JOMFP</journal-id><journal-title-group><journal-title>Journal of Oral and Maxillofacial Pathology : JOMFP</journal-title></journal-title-group><issn pub-type="ppub">0973-029X</issn><issn pub-type="epub">1998-393X</issn><publisher><publisher-name>Medknow Publications</publisher-name><publisher-loc>India</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">22529589</article-id><article-id pub-id-type="pmc">3329690</article-id><article-id pub-id-type="publisher-id">JOMFP-15-236</article-id><article-id pub-id-type="doi">10.4103/0973-029X.84513</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Recurrent unicystic ameloblastoma in a child</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Ponniah</surname><given-names>Irulandy</given-names></name><xref ref-type="aff" rid="aff1"></xref><xref ref-type="corresp" rid="cor1"></xref></contrib></contrib-group><aff id="aff1"><italic>Department of Oral and Maxillofacial Pathology, Tamil Nadu Government Dental College and Hospital, Chennai, India</italic></aff><author-notes><corresp id="cor1"><bold>Address for correspondence:</bold><italic>Dr. Irulandy Ponniah, Department of Oral and Maxillofacial Pathology, Tamil Nadu Government Dental College and Hospital, Chennai-600 003, India. E-mail: <email xlink:href="salivaryduct@yahoo.co.uk">salivaryduct@yahoo.co.uk</email></italic></corresp></author-notes><pub-date pub-type="ppub"><season>May-Aug</season><year>2011</year></pub-date><volume>15</volume><issue>2</issue><fpage>236</fpage><lpage>238</lpage><permissions><copyright-statement>Copyright: © Journal of Oral and Maxillofacial Pathology</copyright-statement><copyright-year>2011</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>Unicystic ameloblastoma (UCA) is a clinical subtype of ameloblastoma that is considered prognostically different. The purpose of this report is to present a case of UCA showing dual radiographic pattern in a child. A detailed study of the lesion was carried out in an 8 year old female child who presented to our department of oral and maxillofacial pathology. Clinical, radiological and histopathological findings were recorded. In March 2005, a painless swelling in the left side of the mandible was noted, which on radiographic examination showed a unilocular radioluceny enclosing the crown of mandibular left permanent second molar, extending between the left first permanent molar and anterior margin of the ramus. Histopathologic diagnosis was UCA. The lesion was treated by enucleation. The patient returned with recurrence in 2009, at this time the lesion radiographically presented as a multilocular radiolucency with a soap bubble appearance, extending between the anterior border of the ramus and second premolar. Histopathologic diagnosis was UCA. The lesion was treated by segmental resection with immediate reconstruction. Although a number of treatment modalities are available to treat UCA, many factors need to be taken into consideration in the treatment of UCA in children.</p></abstract><kwd-group><kwd>Multilocular</kwd><kwd>second molar</kwd><kwd>unilocular</kwd><kwd>unicystic ameloblastoma</kwd></kwd-group></article-meta></front><body><sec id="sec1-1"><title>INTRODUCTION</title><p>Unicystic ameloblastoma (UCA) was first described by Robinson and Martinez,[<xref ref-type="bibr" rid="ref1">1</xref>] to delineate its less aggressive course compared to solid ameloblastoma. With the exception of age of onset and biologic outcome,[<xref ref-type="bibr" rid="ref1">1</xref><xref ref-type="bibr" rid="ref2">2</xref>] this entity does not differ much from the solid ameloblastoma.[<xref ref-type="bibr" rid="ref2">2</xref>] The purpose of this report is to present a case of UCA showing a pericoronal radiolucency in the primary lesion and multilocular radiolucency in the recurrent lesion.</p></sec><sec id="sec1-2"><title>CASE REPORT</title><p>An 8-year old girl presented in March 2005, with a painless swelling on the left side of the mandible of 1 month duration. Extra orally, slight asymmetry was noted and intra orally, the swelling was seen extending from mandibular left permanent first molar to the retro molar region. Panoramic radiograph showed a unilocular radiolucency between mandibular left first permanent molar and anterior margin of the ramus, enclosing the crown of permanent second molar [<xref ref-type="fig" rid="F1">Figure 1</xref>]. Aspiration showed positive results. Clinical provisional diagnosis was dentigerous cyst.</p><fig id="F1" position="float"><label>Figure 1</label><caption><p>Panoramic radiographic examination of the initial lesion showing a unilocular radiolucency between mandibular left first permanent molar and anterior margin of the ramus, enclosing the crown of the permanent second molar</p></caption><graphic xlink:href="JOMFP-15-236-g001"></graphic></fig><p>Histologically, the incisional specimen was characterized by a cystic lining with ameloblast-like basal cells and stellate reticulum-like overlying cells. The fibrous wall was devoid of odontogenic islands and slightly myxoid in character [<xref ref-type="fig" rid="F2">Figure 2</xref>]. In April 2005, the lesion was enucleated and a single cystic sac was noted. The post surgical specimen showed same features as incisional biopsy specimen but the fibrous wall showed inactive odontogenic islands [<xref ref-type="fig" rid="F2">Figure 2</xref>-Inset]. The lesion was typed as simple unicystic ameloblastoma. She was lost to follow-up.</p><fig id="F2" position="float"><label>Figure 2</label><caption><p>Histopathological examination of the incisional specimen characterized by a cystic lining ameloblast-like basal cells and stellate reticulum-like overlying cells. The fibrous wall is slightly myxoid in character. INSET – fibrous wall showing inactive odontogenic rests (H and E stain, 60×; 240×)</p></caption><graphic xlink:href="JOMFP-15-236-g002"></graphic></fig><p>The patient returned with the complaints of pain and swelling in 2009. Extra orally, mild asymmetry was noted on the left side of the mandible. It was hard and tender to palpation. Intra orally, the swelling was found in the edentulous region in relation to missing second permanent molar. Radiographically (panoramic and lateral oblique view radiographs), a multiculocular, soap bubble, radiolucency was found between the anterior border of the ramus and second premolar [Figures <xref ref-type="fig" rid="F3">3</xref> and <xref ref-type="fig" rid="F4">4</xref>]. Incisional biopsy revealed a cystic lesion consistent with unicystic ameloblastoma. The lesion was treated by segmental resection with immediate reconstruction using a low cost stainless steel mandibular plate [<xref ref-type="fig" rid="F5">Figure 5</xref>]. Post surgical evaluation showed a monocystic lesion with expanded but intact cortical plate. The lower border showed no perforation [<xref ref-type="fig" rid="F6">Figure 6a</xref>]. Microscopically, an epithelial lining with intraluminal plexiform growth was observed [<xref ref-type="fig" rid="F6">Figure 6b</xref>]. Multiple blocks (soft and hard tissues) and serial sections were evaluated; however, neither mural component nor infiltration into bony margins was seen.</p><fig id="F3" position="float"><label>Figure 3</label><caption><p>Panoramic radiographic examination of the recurrent lesion showing a multilocular, soap bubble, radioluceny between the anterior border of the left ramus and left second premolar</p></caption><graphic xlink:href="JOMFP-15-236-g003"></graphic></fig><fig id="F4" position="float"><label>Figure 4</label><caption><p>Lateral oblique view radiograph of the recurrent lesion showing a multilocular appearance between the anterior border of the left ramus and left second premolar</p></caption><graphic xlink:href="JOMFP-15-236-g004"></graphic></fig><fig id="F5" position="float"><label>Figure 5</label><caption><p>Low cost stainless steel mandibular reconstruction plate fixed immediately after mandibular segmental resection</p></caption><graphic xlink:href="JOMFP-15-236-g005"></graphic></fig><fig id="F6" position="float"><label>Figure 6</label><caption><p>(a) Post surgical radiographic evaluation showing a monocystic lesion with expanded but intact cortical plate and no lower border peroforation. (b) Histopathologic examination of a specimen of the recurrent lesion showing an epithelial lining with intraluminal plexiform (H and E stain, 40×)</p></caption><graphic xlink:href="JOMFP-15-236-g006"></graphic></fig></sec><sec id="sec1-3"><title>DISCUSSION</title><p>UCA, strictly, implies not a unilocular radiographic pattern but refers to the monocystic appearance, grossly and microscopically.[<xref ref-type="bibr" rid="ref2">2</xref>] Radiographically, UCA can present unilocular, pericoronal and multilocular patterns, each with different age predilection.[<xref ref-type="bibr" rid="ref2">2</xref>] In the current case, a pericoronal radiolucency was found 4 years earlier (primary) and a multilocular radiolucency 4 years later (recurrent). There is no available information in the literature about the dual radiographic pattern between the primary and the recurrent lesion. In Robinson and Martinez,[<xref ref-type="bibr" rid="ref1">1</xref>] series, the mean age for dentigerous radiographic pattern was 19 years and 47 years for non-dentigerous unilocular lesions. On the other hand, Eversole <italic>et al</italic>,[<xref ref-type="bibr" rid="ref3">3</xref>] found a mean age of 33 years for multilocular and 19 years for lesions with a unilocular radiographic pattern. These indicate that UCA may show multilocular radiographic pattern in older adults rather than children. In the current case, both the primary and recurrent lesions occurred at a lower age. This is not unusual as others have also found UCA in children less than 15 years of age.[<xref ref-type="bibr" rid="ref1">1</xref><xref ref-type="bibr" rid="ref4">4</xref>–<xref ref-type="bibr" rid="ref6">6</xref>] In general, UCA frequently is found in association with mandibular third molars,[<xref ref-type="bibr" rid="ref2">2</xref>] while it is less frequently found in relation to permanent second molar or premolars.[<xref ref-type="bibr" rid="ref1">1</xref><xref ref-type="bibr" rid="ref4">4</xref><xref ref-type="bibr" rid="ref5">5</xref><xref ref-type="bibr" rid="ref7">7</xref>].</p><p>According to Robinson and Martinez,[<xref ref-type="bibr" rid="ref1">1</xref>] UCA may show either ameloblastic type of epithelial lining cells or non-ameloblastomatous epithelium. UCA are typed into three subtypes with simple unicystic, luminal and mural categories[<xref ref-type="bibr" rid="ref8">8</xref>] with implications for difference in biologic behavior.[<xref ref-type="bibr" rid="ref2">2</xref><xref ref-type="bibr" rid="ref8">8</xref><xref ref-type="bibr" rid="ref9">9</xref>]</p><p>There are a number of views regarding the genesis of UCA;[<xref ref-type="bibr" rid="ref2">2</xref>] de novo origin, from non-neoplastic odontogenic cyst and cystic degeneration of solid ameloblastoma. In our case, both primary and the recurrent lesion displayed ameloblastic epithelium and both were of simple UCA type. A similar finding of identical histology of primary and recurrent lesion was also found by Rosenstein <italic>et al</italic>.[<xref ref-type="bibr" rid="ref9">9</xref>]</p><p>Treatment modalities for UCA include enucleation, enucleation followed by application of Carnoy's solution, marsupialization followed by surgery, and resection.[<xref ref-type="bibr" rid="ref10">10</xref>] These surgical techniques offer a success rate of 70%, 82%, 84% and 96%, respectively. Several factors are taken in to consideration in the treatment of UCA in children; these include size, location, duration, mural component, psychological impact, control of possible recurrence and scope for follow-up.[<xref ref-type="bibr" rid="ref4">4</xref>] Psychology plays an important role in the decision making because of its negative impact on growth and function.[<xref ref-type="bibr" rid="ref4">4</xref><xref ref-type="bibr" rid="ref11">11</xref>] Further, the precise histological subtyping of UCA or distinction from solid ameloblastoma is often difficult to make, prior to definitive surgery.[<xref ref-type="bibr" rid="ref12">12</xref>] In view of this, primary lesions are treated by means of conservative procedure; however, when odontogenic cell rests or mural component are found in the fibrous wall, it would be prudent to resect uninvolved normal bone along with lesional tissue.[<xref ref-type="bibr" rid="ref4">4</xref><xref ref-type="bibr" rid="ref8">8</xref>] In our case, odontogenic cell rests were not apparent in the primary lesion and was found only on post surgical evaluation. The short recurrence time in the current case might well be related to the presence of odontogenic rests. The current treatment policy for UCA advocates more aggressive measures, even for primary lesion, such as peripheral osteotomy or segmental resection.[<xref ref-type="bibr" rid="ref13">13</xref>] In conclusion, a case of UCA in a child with a dual radiographic pattern is presented.</p></sec></body><back><fn-group><fn fn-type="supported-by"><p><bold>Source of Support:</bold> Nil</p></fn><fn fn-type="conflict"><p><bold>Conflict of Interest:</bold> None declared.</p></fn></fn-group><ref-list><title>REFERENCES</title><ref id="ref1"><label>1</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Robinson</surname><given-names>L</given-names></name><name><surname>Martinez</surname><given-names>MG</given-names></name></person-group><article-title>A prognostically distinct entity</article-title><source>Cancer</source><year>1977</year><volume>40</volume><fpage>2278</fpage><lpage>85</lpage><pub-id 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