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Acanthomatous Ameloblastoma in Anterior Mandibular Region of a Young Patient: A Rare Case Report

Identifieur interne : 001B25 ( Pmc/Corpus ); précédent : 001B24; suivant : 001B26

Acanthomatous Ameloblastoma in Anterior Mandibular Region of a Young Patient: A Rare Case Report

Auteurs : Sridevi Ugrappa ; Ajay Jain ; Neeraj Kumar Fuloria ; Shivkanya Fuloria

Source :

RBID : PMC:5452712

Abstract

Ameloblastoma is the most known of the epithelial odontogenic benign tumor. It is slow growing and locally aggressive in nature and most commonly seen in the posterior mandible. Various histopathological variants exist, among which acanthomatous type of ameloblastoma is one of the rarest types. Acanthomatous ameloblastoma is usually seen in older aged human population and most commonly reported in canine region of dogs in literature. Here, we report a rare case of acanthomatous ameloblastoma in a young male patient involving mandibular anterior region crossing the midline with recurrence over a period of 2 years of follow-up after surgical resection.


Url:
DOI: 10.4103/aam.aam_51_16
PubMed: 28469123
PubMed Central: 5452712

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PMC:5452712

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<title xml:lang="en">Acanthomatous Ameloblastoma in Anterior Mandibular Region of a Young Patient: A Rare Case Report</title>
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<name sortKey="Ugrappa, Sridevi" sort="Ugrappa, Sridevi" uniqKey="Ugrappa S" first="Sridevi" last="Ugrappa">Sridevi Ugrappa</name>
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<nlm:aff id="aff1">Department of Oral Medicine and Radiology, Faculty of Dentistry, AIMST Dental Institute, AIMST University, Kedah, Malaysia</nlm:aff>
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<name sortKey="Jain, Ajay" sort="Jain, Ajay" uniqKey="Jain A" first="Ajay" last="Jain">Ajay Jain</name>
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<nlm:aff id="aff2">Department of Prosthodontics, Faculty of Dentistry, AIMST Dental Institute, AIMST University, Kedah, Malaysia</nlm:aff>
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<name sortKey="Fuloria, Neeraj Kumar" sort="Fuloria, Neeraj Kumar" uniqKey="Fuloria N" first="Neeraj Kumar" last="Fuloria">Neeraj Kumar Fuloria</name>
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<nlm:aff id="aff3">Pharmaceutical Chemistry Unit, Faculty of Pharmacy, AIMST University, Kedah, Malaysia</nlm:aff>
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<name sortKey="Fuloria, Shivkanya" sort="Fuloria, Shivkanya" uniqKey="Fuloria S" first="Shivkanya" last="Fuloria">Shivkanya Fuloria</name>
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<title xml:lang="en" level="a" type="main">Acanthomatous Ameloblastoma in Anterior Mandibular Region of a Young Patient: A Rare Case Report</title>
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<name sortKey="Jain, Ajay" sort="Jain, Ajay" uniqKey="Jain A" first="Ajay" last="Jain">Ajay Jain</name>
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<name sortKey="Fuloria, Neeraj Kumar" sort="Fuloria, Neeraj Kumar" uniqKey="Fuloria N" first="Neeraj Kumar" last="Fuloria">Neeraj Kumar Fuloria</name>
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<p>Ameloblastoma is the most known of the epithelial odontogenic benign tumor. It is slow growing and locally aggressive in nature and most commonly seen in the posterior mandible. Various histopathological variants exist, among which acanthomatous type of ameloblastoma is one of the rarest types. Acanthomatous ameloblastoma is usually seen in older aged human population and most commonly reported in canine region of dogs in literature. Here, we report a rare case of acanthomatous ameloblastoma in a young male patient involving mandibular anterior region crossing the midline with recurrence over a period of 2 years of follow-up after surgical resection.</p>
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<pmc article-type="case-report">
<pmc-dir>properties open_access</pmc-dir>
<front>
<journal-meta>
<journal-id journal-id-type="nlm-ta">Ann Afr Med</journal-id>
<journal-id journal-id-type="iso-abbrev">Ann Afr Med</journal-id>
<journal-id journal-id-type="publisher-id">AAM</journal-id>
<journal-title-group>
<journal-title>Annals of African Medicine</journal-title>
</journal-title-group>
<issn pub-type="ppub">1596-3519</issn>
<issn pub-type="epub">0975-5764</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">28469123</article-id>
<article-id pub-id-type="pmc">5452712</article-id>
<article-id pub-id-type="publisher-id">AAM-16-85</article-id>
<article-id pub-id-type="doi">10.4103/aam.aam_51_16</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Case Report</subject>
</subj-group>
</article-categories>
<title-group>
<article-title>Acanthomatous Ameloblastoma in Anterior Mandibular Region of a Young Patient: A Rare Case Report</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<name>
<surname>Ugrappa</surname>
<given-names>Sridevi</given-names>
</name>
<xref ref-type="aff" rid="aff1"></xref>
<xref ref-type="corresp" rid="cor1"></xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Jain</surname>
<given-names>Ajay</given-names>
</name>
<xref ref-type="aff" rid="aff2">1</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Fuloria</surname>
<given-names>Neeraj Kumar</given-names>
</name>
<xref ref-type="aff" rid="aff3">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Fuloria</surname>
<given-names>Shivkanya</given-names>
</name>
<xref ref-type="aff" rid="aff3">2</xref>
</contrib>
</contrib-group>
<aff id="aff1">Department of Oral Medicine and Radiology, Faculty of Dentistry, AIMST Dental Institute, AIMST University, Kedah, Malaysia</aff>
<aff id="aff2">
<label>1</label>
Department of Prosthodontics, Faculty of Dentistry, AIMST Dental Institute, AIMST University, Kedah, Malaysia</aff>
<aff id="aff3">
<label>2</label>
Pharmaceutical Chemistry Unit, Faculty of Pharmacy, AIMST University, Kedah, Malaysia</aff>
<author-notes>
<corresp id="cor1">
<bold>Address for correspondence:</bold>
Dr. Sridevi Ugrappa, Department of Oral Medicine and Radiology, Faculty of Dentistry, AIMST Dental Institute, AIMST University, Kedah, Malaysia. E-mail:
<email xlink:href="srideviugrappa@gmail.com">srideviugrappa@gmail.com</email>
</corresp>
</author-notes>
<pub-date pub-type="ppub">
<season>Apr-Jun</season>
<year>2017</year>
</pub-date>
<volume>16</volume>
<issue>2</issue>
<fpage>85</fpage>
<lpage>89</lpage>
<permissions>
<copyright-statement>Copyright: © 2017 Annals of African Medicine</copyright-statement>
<copyright-year>2017</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>Ameloblastoma is the most known of the epithelial odontogenic benign tumor. It is slow growing and locally aggressive in nature and most commonly seen in the posterior mandible. Various histopathological variants exist, among which acanthomatous type of ameloblastoma is one of the rarest types. Acanthomatous ameloblastoma is usually seen in older aged human population and most commonly reported in canine region of dogs in literature. Here, we report a rare case of acanthomatous ameloblastoma in a young male patient involving mandibular anterior region crossing the midline with recurrence over a period of 2 years of follow-up after surgical resection.</p>
</abstract>
<trans-abstract xml:lang="fr">
<title>Résumé</title>
<p>L’améloblastome est la plus connue de la tumeur bénigne odontogénique épithéliale. Il est de croissance lente et de nature localement agressif et la plupart Communément vu dans la mandibule postérieure. Diverses variantes histopathologiques existent, dont le type d’améloblastome acanthomateux est L’un des types les plus rares. L’ameloblastome acanthomateux est habituellement observé chez les personnes âgées âgées et le plus souvent signalé chez les canins Région des chiens en littérature. Ici, nous signalons un cas rare d’ameloblastome acanthomateux chez un jeune malade impliquant un antérieur mandibulaire Région traversant la ligne médiane avec récurrence sur une période de 2 ans de suivi après résection chirurgicale.</p>
</trans-abstract>
<kwd-group>
<kwd>Acanthomatous ameloblastoma</kwd>
<kwd>benign odontogenic tumor</kwd>
<kwd>recurrence</kwd>
<kwd>Ameloblastome acanthomateux</kwd>
<kwd>récurrence</kwd>
<kwd>tumeur odontogène bénigne</kwd>
</kwd-group>
</article-meta>
</front>
<body>
<sec sec-type="intro" id="sec1-1">
<title>I
<sc>ntroduction</sc>
</title>
<p>Many benign tumors of the orofacial region cause swellings which are either of odontogenic or nonodontogenic in origin. Of many, the most common benign odontogenic tumor is ameloblastoma and is described for the first time by Broca in 1868 as adamantinoma and then recoined by Churchill in 1934.[
<xref rid="ref1" ref-type="bibr">1</xref>
] Higher incidence of occurrence is reported in the mandibular molar-ramus region of about 70% than maxilla by the ratio of 5:1, and it accounts for 1% of all cysts/tumors of jaws and 18% of all odontogenic neoplasms.[
<xref rid="ref2" ref-type="bibr">2</xref>
] Average age reported is in the third to fifth decade of life with no sex predilection when the tumor is not associated with an unerupted tooth; the gender ratio is male to female ratio of 1:1.8.[
<xref rid="ref3" ref-type="bibr">3</xref>
] According to the current World Health Organization classification of odontogenic tumors, ameloblastomas are divided into four types: solid/multicystic, extraosseous/peripheral, desmoplastic, and unicystic. Various histologically subtypes have been described, including those of follicular, plexiform, acanthomatous, granular, and basal cells. Most literatures showed that follicular ameloblastoma is the most prevalent histological variant (64.9%) followed by the plexiform (13.0%), desmoplastic (5.2%), and acanthomatous (3.9%) varieties.[
<xref rid="ref4" ref-type="bibr">4</xref>
<xref rid="ref5" ref-type="bibr">5</xref>
] This case report presents a distinctive occurrence of acanthomatous ameloblastoma in a young male patient crossing the midline of the anterior mandible, which is the rarest case reported.</p>
</sec>
<sec id="sec1-2">
<title>C
<sc>ase</sc>
R
<sc>eport</sc>
</title>
<p>An 18-year-old male patient reported to the Department of Oral Medicine and Radiology with a chief complaint of swelling in his lower front region of the jaw for 4 months. The patient gave a history of swelling which is gradually increasing in size to the present. No history of pain, trauma, or any discharge from the swelling was elucidated.</p>
<p>On extraoral examination, a diffuse swelling was seen over the chin region extending from the left angle of mouth crossing midline to the right of the angle of mouth, measuring approximately 4 cm × 3 cm in size with obliteration of the mentolabial sulcus [
<xref ref-type="fig" rid="F1">Figure 1</xref>
]. On palpation, swelling was nontender, with movable overlying skin, soft in consistency, and crepitus was felt over the chin region.</p>
<fig id="F1" position="float">
<label>Figure 1</label>
<caption>
<p>Extraoral preoperative photograph</p>
</caption>
<graphic xlink:href="AAM-16-85-g001"></graphic>
</fig>
<p>On intraoral examination, obliteration of the lower labial vestibule and alveolingual sulcus extending from teeth 36 to 45, crossing the midline and superior-inferiorly from the attached gingiva to depth of the labial and buccal vestibule measuring approximately 5 cm × 3 cm in size. Displacement of the teeth anteriomesially in relation to 41, 42 and anteriodistally in relation to 31 was seen [
<xref ref-type="fig" rid="F2">Figure 2</xref>
]. On palpation, crepitus was felt on labial vestibule in relation to teeth 34, 35, 33, 32, 42, 43, 44, and 45 with nontender swelling and soft in consistency.</p>
<fig id="F2" position="float">
<label>Figure 2</label>
<caption>
<p>Intraoral view</p>
</caption>
<graphic xlink:href="AAM-16-85-g002"></graphic>
</fig>
<p>Pulp vitality test was performed in relation to teeth 35–45 and delayed response was noted. A clear yellow-colored fluid was obtained upon aspiration [
<xref ref-type="fig" rid="F3">Figure 3</xref>
]. Based on patient history, clinical examination, and chairside investigations, benign odontogenic tumor was considered as provisional diagnosis. Differential diagnosis includes radicular cyst, ameloblastoma, odontogenic keratocyst, central giant cell granuloma, and odontogenic myxoma.</p>
<fig id="F3" position="float">
<label>Figure 3</label>
<caption>
<p>Yellow-colored clear aspiration fluid</p>
</caption>
<graphic xlink:href="AAM-16-85-g003"></graphic>
</fig>
<p>Further investigations were performed such as orthopantomograph (OPG), occlusal anterior mandibular view, computed tomography (CT) scan, and incisional biopsy. OPG interpretation revealed well-defined multilocular radiolucent area with corticated borders and internal septa giving appearance of soap bubble extending from the mesial root of tooth 35 to mesial root of tooth 45 involving midline with root resorption of apical one-third in relation to teeth 33, 32, 31, 41, 42, and 43 and displacement of teeth 31 and 32 [
<xref ref-type="fig" rid="F4">Figure 4</xref>
]. Anterior occlusal mandibular view revealed expansion of the buccal/labial and lingual cortical plates from the tooth 36 region crossing the midline up to the tooth 46 region, with the presence internal septa in relation to teeth 46, 45, 44, and 43 regions and with a very thin corticated boundary [
<xref ref-type="fig" rid="F5">Figure 5</xref>
]. CT scan showed an expansile osteolytic radiolucent lesion in the anterior mandible, with expansion and thinning of lingual cortical plate and expansion and break in the continuity of labial cortical plate [Figures
<xref ref-type="fig" rid="F6">6</xref>
<xref ref-type="fig" rid="F8">8</xref>
]. Radiological differential diagnosis includes ameloblastoma, central giant cell granuloma, and odontogenic myxoma.</p>
<fig id="F4" position="float">
<label>Figure 4</label>
<caption>
<p>Orthopantomograph</p>
</caption>
<graphic xlink:href="AAM-16-85-g004"></graphic>
</fig>
<fig id="F5" position="float">
<label>Figure 5</label>
<caption>
<p>Anterior mandibular occlusal view</p>
</caption>
<graphic xlink:href="AAM-16-85-g005"></graphic>
</fig>
<fig id="F6" position="float">
<label>Figure 6</label>
<caption>
<p>Axial view of computed tomography scan</p>
</caption>
<graphic xlink:href="AAM-16-85-g006"></graphic>
</fig>
<fig id="F7" position="float">
<label>Figure 7</label>
<caption>
<p>Coronal view of computed tomography scan</p>
</caption>
<graphic xlink:href="AAM-16-85-g007"></graphic>
</fig>
<fig id="F8" position="float">
<label>Figure 8</label>
<caption>
<p>Three-dimensional computed tomography scan</p>
</caption>
<graphic xlink:href="AAM-16-85-g008"></graphic>
</fig>
<p>Incisional biopsy was performed later [
<xref ref-type="fig" rid="F9">Figure 9</xref>
], and histopathological features showed periphery of the follicle which was lined by a single layer of tall columnar ameloblast like cells, and central region showed loosely arranged polygonal or angular cells resembling stellate reticulum. Many solid epithelial cell nests also showed squamous differentiation with well-formed keratin pearls suggestive of acanthomatous ameloblastoma [
<xref ref-type="fig" rid="F10">Figure 10</xref>
].</p>
<fig id="F9" position="float">
<label>Figure 9</label>
<caption>
<p>Incisional biopsy</p>
</caption>
<graphic xlink:href="AAM-16-85-g009"></graphic>
</fig>
<fig id="F10" position="float">
<label>Figure 10</label>
<caption>
<p>Histopathology</p>
</caption>
<graphic xlink:href="AAM-16-85-g010"></graphic>
</fig>
<p>After obtaining the consent from the patient, surgical resection of the anterior mandible with wide normal margins followed by reconstruction with iliac cancellous bone was done. Satisfactory postoperative healing was noted, and replacement with fixed-removable prosthesis was delivered to the patient [Figures
<xref ref-type="fig" rid="F11">11</xref>
<xref ref-type="fig" rid="F13">13</xref>
]. After 2 years of follow-up period, OPG and intraoral periapical radiograph [Figure
<xref ref-type="fig" rid="F14">14a</xref>
and
<xref ref-type="fig" rid="F14">b</xref>
] were taken, which revealed satisfactory healing of the resected area. However, a well-defined radiolucency in the edentulous region adjacent to the tooth 34 was noted, which may be a sign of recurrence and was scheduled for further treatment.</p>
<fig id="F11" position="float">
<label>Figure 11</label>
<caption>
<p>Postoperative follow-up intraoral view</p>
</caption>
<graphic xlink:href="AAM-16-85-g011"></graphic>
</fig>
<fig id="F12" position="float">
<label>Figure 12</label>
<caption>
<p>Postoperative follow-up with fixed-removable prosthesis</p>
</caption>
<graphic xlink:href="AAM-16-85-g012"></graphic>
</fig>
<fig id="F13" position="float">
<label>Figure 13</label>
<caption>
<p>Postoperative orthopantomograph</p>
</caption>
<graphic xlink:href="AAM-16-85-g013"></graphic>
</fig>
<fig id="F14" position="float">
<label>Figure 14</label>
<caption>
<p>(a and b) Two-year follow-up orthopantomograph and intraoral periapical radiograph</p>
</caption>
<graphic xlink:href="AAM-16-85-g014"></graphic>
</fig>
</sec>
<sec sec-type="discussion" id="sec1-3">
<title>D
<sc>iscussion</sc>
</title>
<p>Ameloblastoma is usually asymptomatic and found on routine dental X-rays; however, they present with jaw expansion. It is slow but relentless growth may cause movement of tooth roots or root resorption as seen in our case, loose teeth, malocclusion, or more rarely paresthesia and pain.[
<xref rid="ref6" ref-type="bibr">6</xref>
]</p>
<p>Average age reported is in the third to fifth decade of life, whereas acanthomatous ameloblastoma as reported to occur mostly in the seventh decade of life.[
<xref rid="ref7" ref-type="bibr">7</xref>
<xref rid="ref5" ref-type="bibr">5</xref>
] Unlike the present case, the most common site of occurrence is in the mandible (80% of ameloblastomas) with 70% are located in the area of the molars or the ascending ramus,[
<xref rid="ref7" ref-type="bibr">7</xref>
<xref rid="ref8" ref-type="bibr">8</xref>
] whereas the present case was reported, with occurrence in the mandibular anterior region crossing midline and displacing anterior teeth, which is the rarest reported human case in literature.</p>
<p>Radiographic features of ameloblastoma most commonly appear as radiolucent, unilocular/multiloculated cystic lesion, with a characteristic “soap bubble-like” appearance, cortical thinning/destruction with local invasion, and root resorptions.[
<xref rid="ref3" ref-type="bibr">3</xref>
] These characteristic radiographic features are evident in our present case.</p>
<p>Ameloblastomas have been classified in both human and veterinary literature[
<xref rid="ref9" ref-type="bibr">9</xref>
] and have been defined as benign, locally invasive, and clinically malignant lesions. Metastasis has been documented only in humans as malignant ameloblastomas and ameloblastic carcinomas have been noted to metastasize to the lungs, pleura, orbit, skull, and brain but not in dogs.[
<xref rid="ref9" ref-type="bibr">9</xref>
] Adebiyi
<italic>et al</italic>
. reported histopathological types that the most of the solid lesions belonged to follicular ameloblastoma (64.9%) followed by the plexiform (13.0%), desmoplastic (5.2%), and acanthomatous (3.9%).[
<xref rid="ref5" ref-type="bibr">5</xref>
] Histopathologically, acanthomatous type shows central squamous cell differential with keratin formation as seen in the present case. Some authors stated that formation of squamous metaplasia may be due to chronic irritation of calculus and oral sepsis.[
<xref rid="ref10" ref-type="bibr">10</xref>
] Treatment modality varies depending on the type of ameloblastoma diagnosed as unicystic/multicystic.[
<xref rid="ref8" ref-type="bibr">8</xref>
<xref rid="ref11" ref-type="bibr">11</xref>
] Surgical resection is the treatment of choice for most of the solid/multicystic ameloblastomas. The recurrence rate reported in the literature is 13%–15% for surgical resection and 90%–100% for curettage.[
<xref rid="ref8" ref-type="bibr">8</xref>
] Follow-up of the patient is utmost essential as the chance of recurrences present within the first 5 years,[
<xref rid="ref11" ref-type="bibr">11</xref>
] which is as seen in our case after a period of 2 years. Some authors also believe that high chance of the acanthomatous variant turning into metastasizing squamous cell carcinoma, if left untreated. There are controversies about the biological behavior of the acanthomatous ameloblastoma; some researchers believe that it is locally aggressive and frequently invades the alveolar bone or recurs after marginal surgical excision as it was reported in our present case. Some others believe that there is no difference among the various subtypes of ameloblastoma.[
<xref rid="ref12" ref-type="bibr">12</xref>
<xref rid="ref13" ref-type="bibr">13</xref>
] Thus, the study of molecular mechanisms of cell proliferation can be helpful to predict the aggressiveness of ameloblastoma and especially p16 (cyclin-dependent kinase inhibitor) which is a tumoral suppressor protein encoded by the CDKN2A gene. A high score in the immunoexpression of the p16 protein may indicate lower aggressiveness and a lower rate of recurrence.[
<xref rid="ref14" ref-type="bibr">14</xref>
]</p>
</sec>
<sec sec-type="conclusion" id="sec1-4">
<title>C
<sc>onclusion</sc>
</title>
<p>Although ameloblastoma is a most common benign tumor, the incidence of occurrence of acanthomatous ameloblastoma is very rare in human beings. To our knowledge, very few cases of acanthomatous type of ameloblastomas are reported in elderly aged patients occuring in mandibular posterior region, and cases of this type of ameloblastomas in younger aged patients (second decade) occurring in the anterior mandibular region crossing midline with recurrence over a period of time are not reported in the literature. Hence, further study of molecular mechanism and implication in clinical practice has to be considered.</p>
<sec id="sec2-1">
<title>Declaration of patient consent</title>
<p>The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.</p>
</sec>
<sec id="sec2-2">
<title>Financial support and sponsorship</title>
<p>Nil.</p>
</sec>
<sec id="sec2-3" sec-type="COI-statement">
<title>Conflicts of interest</title>
<p>There are no conflicts of interest.</p>
</sec>
</sec>
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