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<title xml:lang="en">Solitary peripheral osteomas of the jaws</title>
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<name sortKey="De Franca, Talita Ribeiro Ten Rio" sort="De Franca, Talita Ribeiro Ten Rio" uniqKey="De Franca T" first="Talita Ribeiro Ten Rio" last="De França">Talita Ribeiro Ten Rio De França</name>
<affiliation>
<nlm:aff id="A1">Department of Clinical and Preventive Dentistry, Oral Pathology Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Gueiros, Luiz Alcino Monteiro" sort="Gueiros, Luiz Alcino Monteiro" uniqKey="Gueiros L" first="Luiz Alcino Monteiro" last="Gueiros">Luiz Alcino Monteiro Gueiros</name>
<affiliation>
<nlm:aff id="A2">Department of Clinical and Preventive Dentistry, Oral Medicine Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
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<author>
<name sortKey="De Castro, Jurema Freire Lisboa" sort="De Castro, Jurema Freire Lisboa" uniqKey="De Castro J" first="Jurema Freire Lisboa" last="De Castro">Jurema Freire Lisboa De Castro</name>
<affiliation>
<nlm:aff id="A1">Department of Clinical and Preventive Dentistry, Oral Pathology Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Catunda, Ivson" sort="Catunda, Ivson" uniqKey="Catunda I" first="Ivson" last="Catunda">Ivson Catunda</name>
<affiliation>
<nlm:aff id="A3">Department of Oral and Maxillofacial Surgery, Hospital Geral de Areas, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Leao, Jair Carneiro" sort="Leao, Jair Carneiro" uniqKey="Leao J" first="Jair Carneiro" last="Leão">Jair Carneiro Leão</name>
<affiliation>
<nlm:aff id="A2">Department of Clinical and Preventive Dentistry, Oral Medicine Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Da Cruz Perez, Danyel Elias" sort="Da Cruz Perez, Danyel Elias" uniqKey="Da Cruz Perez D" first="Danyel Elias" last="Da Cruz Perez">Danyel Elias Da Cruz Perez</name>
<affiliation>
<nlm:aff id="A2">Department of Clinical and Preventive Dentistry, Oral Medicine Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
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<title xml:lang="en" level="a" type="main">Solitary peripheral osteomas of the jaws</title>
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<name sortKey="De Franca, Talita Ribeiro Ten Rio" sort="De Franca, Talita Ribeiro Ten Rio" uniqKey="De Franca T" first="Talita Ribeiro Ten Rio" last="De França">Talita Ribeiro Ten Rio De França</name>
<affiliation>
<nlm:aff id="A1">Department of Clinical and Preventive Dentistry, Oral Pathology Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Gueiros, Luiz Alcino Monteiro" sort="Gueiros, Luiz Alcino Monteiro" uniqKey="Gueiros L" first="Luiz Alcino Monteiro" last="Gueiros">Luiz Alcino Monteiro Gueiros</name>
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<nlm:aff id="A2">Department of Clinical and Preventive Dentistry, Oral Medicine Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
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<author>
<name sortKey="De Castro, Jurema Freire Lisboa" sort="De Castro, Jurema Freire Lisboa" uniqKey="De Castro J" first="Jurema Freire Lisboa" last="De Castro">Jurema Freire Lisboa De Castro</name>
<affiliation>
<nlm:aff id="A1">Department of Clinical and Preventive Dentistry, Oral Pathology Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Catunda, Ivson" sort="Catunda, Ivson" uniqKey="Catunda I" first="Ivson" last="Catunda">Ivson Catunda</name>
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<nlm:aff id="A3">Department of Oral and Maxillofacial Surgery, Hospital Geral de Areas, Recife, Pernambuco, Brazil.</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Leao, Jair Carneiro" sort="Leao, Jair Carneiro" uniqKey="Leao J" first="Jair Carneiro" last="Leão">Jair Carneiro Leão</name>
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<nlm:aff id="A2">Department of Clinical and Preventive Dentistry, Oral Medicine Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
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<author>
<name sortKey="Da Cruz Perez, Danyel Elias" sort="Da Cruz Perez, Danyel Elias" uniqKey="Da Cruz Perez D" first="Danyel Elias" last="Da Cruz Perez">Danyel Elias Da Cruz Perez</name>
<affiliation>
<nlm:aff id="A2">Department of Clinical and Preventive Dentistry, Oral Medicine Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</nlm:aff>
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<title level="j">Imaging Science in Dentistry</title>
<idno type="ISSN">2233-7822</idno>
<idno type="eISSN">2233-7830</idno>
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<date when="2012">2012</date>
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<p>Osteoma is a benign osteogenic tumor composed of cancellous or compact bone, classified as peripheral, central, or extraskeletal. Peripheral osteomas are uncommon. Excluding the maxillary sinuses, the maxilla is a rare site for osteomas. The purpose of this report was to describe clinicopathological and radiological features of two peripheral osteomas occurring in the jaws, one located in the mandible and another in the edentulous maxillary alveolar ridge. The tumors were asymptomatic and were fully excised without any complications or recurrence. The lesions were submitted to histopathological analysis and diagnosed as peripheral osteoma, compact type.</p>
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<pmc-dir>properties open_access</pmc-dir>
<front>
<journal-meta>
<journal-id journal-id-type="nlm-ta">Imaging Sci Dent</journal-id>
<journal-id journal-id-type="iso-abbrev">Imaging Sci Dent</journal-id>
<journal-id journal-id-type="publisher-id">ISD</journal-id>
<journal-title-group>
<journal-title>Imaging Science in Dentistry</journal-title>
</journal-title-group>
<issn pub-type="ppub">2233-7822</issn>
<issn pub-type="epub">2233-7830</issn>
<publisher>
<publisher-name>Korean Academy of Oral and Maxillofacial Radiology</publisher-name>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="pmid">22783479</article-id>
<article-id pub-id-type="pmc">3389057</article-id>
<article-id pub-id-type="doi">10.5624/isd.2012.42.2.99</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Case Report</subject>
</subj-group>
</article-categories>
<title-group>
<article-title>Solitary peripheral osteomas of the jaws</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<name>
<surname>de França</surname>
<given-names>Talita Ribeiro Tenório</given-names>
</name>
<xref ref-type="aff" rid="A1"></xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Gueiros</surname>
<given-names>Luiz Alcino Monteiro</given-names>
</name>
<xref ref-type="aff" rid="A2">*</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>de Castro</surname>
<given-names>Jurema Freire Lisboa</given-names>
</name>
<xref ref-type="aff" rid="A1"></xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Catunda</surname>
<given-names>Ivson</given-names>
</name>
<xref ref-type="aff" rid="A3">**</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Leão</surname>
<given-names>Jair Carneiro</given-names>
</name>
<xref ref-type="aff" rid="A2">*</xref>
</contrib>
<contrib contrib-type="author" corresp="yes">
<name>
<surname>da Cruz Perez</surname>
<given-names>Danyel Elias</given-names>
</name>
<xref ref-type="aff" rid="A2">*</xref>
</contrib>
</contrib-group>
<aff id="A1">Department of Clinical and Preventive Dentistry, Oral Pathology Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</aff>
<aff id="A2">
<label>*</label>
Department of Clinical and Preventive Dentistry, Oral Medicine Unit, Federal University of Pernambuco, Recife, Pernambuco, Brazil.</aff>
<aff id="A3">
<label>**</label>
Department of Oral and Maxillofacial Surgery, Hospital Geral de Areas, Recife, Pernambuco, Brazil.</aff>
<author-notes>
<corresp>Correspondence to: Dr. Danyel Elias da Cruz Perez. Department of Clinic and Preventive Dentistry, Oral Pathology Unit, Federal University of Pernambuco, Av. Prof. Moraes Rego, 1235, Cidade Universitária, CEP: 50670-901, Recife/PE, Brazil. Tel) 55-81-2126-7510, Fax) 55-81-2126-8338,
<email>danyel.perez@ufpe.br</email>
</corresp>
</author-notes>
<pub-date pub-type="ppub">
<month>6</month>
<year>2012</year>
</pub-date>
<pub-date pub-type="epub">
<day>25</day>
<month>6</month>
<year>2012</year>
</pub-date>
<volume>42</volume>
<issue>2</issue>
<fpage>99</fpage>
<lpage>103</lpage>
<history>
<date date-type="received">
<day>21</day>
<month>1</month>
<year>2012</year>
</date>
<date date-type="rev-recd">
<day>06</day>
<month>4</month>
<year>2012</year>
</date>
<date date-type="accepted">
<day>13</day>
<month>4</month>
<year>2012</year>
</date>
</history>
<permissions>
<copyright-statement>Copyright © 2012 by Korean Academy of Oral and Maxillofacial Radiology</copyright-statement>
<copyright-year>2012</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0">
<license-p>This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (
<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by-nc/3.0">http://creativecommons.org/licenses/by-nc/3.0</ext-link>
) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
</license>
</permissions>
<abstract>
<p>Osteoma is a benign osteogenic tumor composed of cancellous or compact bone, classified as peripheral, central, or extraskeletal. Peripheral osteomas are uncommon. Excluding the maxillary sinuses, the maxilla is a rare site for osteomas. The purpose of this report was to describe clinicopathological and radiological features of two peripheral osteomas occurring in the jaws, one located in the mandible and another in the edentulous maxillary alveolar ridge. The tumors were asymptomatic and were fully excised without any complications or recurrence. The lesions were submitted to histopathological analysis and diagnosed as peripheral osteoma, compact type.</p>
</abstract>
<kwd-group>
<kwd>Osteoma</kwd>
<kwd>Mandible</kwd>
<kwd>Maxilla</kwd>
</kwd-group>
</article-meta>
</front>
<body>
<p>Osteomas are benign osteogenic tumors characterized by the proliferation of compact and/or cancellous bone.
<xref ref-type="bibr" rid="B1">1</xref>
-
<xref ref-type="bibr" rid="B3">3</xref>
They are essentially tumors of the craniofacial bones, rarely affecting the extragnathic skeleton.
<xref ref-type="bibr" rid="B4">4</xref>
The osteomas may be central, peripheral, or extraskeletal. The central type arises from the endosteum, the peripheral counterpart from the periosteum, and the extraskeletal soft tissue osteomas usually develop within muscles.
<xref ref-type="bibr" rid="B4">4</xref>
-
<xref ref-type="bibr" rid="B6">6</xref>
</p>
<p>The reported age at onset ranges from 14 to 58 years, with a mean of 29 years of age, and males are affected more frequently than females in a ratio of approximately 2 : 1. They mainly affect the frontal bone, mandible, or paranasal sinuses.
<xref ref-type="bibr" rid="B1">1</xref>
Excluding the maxillary sinuses and those cases associated with Gardner syndrome, maxillary osteomas are rare.
<xref ref-type="bibr" rid="B3">3</xref>
Recently, Nah
<xref ref-type="bibr" rid="B7">7</xref>
reported 18 cases of craniofacial osteomas and only 1 case occurred in the maxilla. To the best of our knowledge, only 17 well-documented cases of maxillary osteomas have been previously described in the English-language literature, 13 peripheral and 4 central (
<xref ref-type="table" rid="T1">Table 1</xref>
).
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B3">3</xref>
,
<xref ref-type="bibr" rid="B7">7</xref>
-
<xref ref-type="bibr" rid="B17">17</xref>
Thus, this report described the clinicopathological and radiological features of two solitary peripheral osteomas of the jaws, one located on the mandible and the other on the edentulous maxillary alveolar ridge.</p>
<sec>
<title>Case Reports</title>
<sec>
<title>Case 1</title>
<p>A 33-year-old female patient presented to the Oral Surgery Clinic, complaining of a painless mandibular swelling with approximately 5 years of evolution. The medical history was not contributory. On extraoral examination, a nodule with a lobulated surface, of bony consistency, fixed, extending 2.5 cm, and located on the mandibular angle was observed. The entire tumor was covered by normal skin. Intraorally, no alteration was observed. The computed tomography (CT) scan showed a well-circumscribed, bone-like, hyperdense image (
<xref ref-type="fig" rid="F1">Fig. 1A</xref>
) with a lobulated surface located on the left mandible angle (
<xref ref-type="fig" rid="F1">Fig. 1B</xref>
). Peripheral osteoma was the clinicoradiographic diagnosis. Under general anesthesia, the lesion was fully excised, and histopathological analysis showed compact, mature bone, with scarce lacunae and marrow spaces filled by connective tissue within the compact bone (
<xref ref-type="fig" rid="F2">Fig. 2</xref>
). Based upon the clinical and laboratory findings, a definitive diagnosis of peripheral osteoma, compact type, was established. The patient did not present postoperative complications nor clinical or imaging signs of recurrence after 8 months of follow up.</p>
</sec>
<sec>
<title>Case 2</title>
<p>A 53-year-old male patient was referred to the Oral Medicine Unit for evaluation of a mass in the posterior maxilla originating about 10 years earlier. The patient had good general health with no history of contributory medical factors. Intraoral examination revealed a painless, well-circumscribed, submucosal mass on the right edentulous posterior maxillary alveolar ridge, covered by regular and normally colored mucosa, measuring approximately 1.0 cm in diameter. The lesion presented a hard consistency on palpation, and it was located on the buccal aspect of the maxilla, in the transition line between the attached mucosa and the mobile alveolar mucosa. The nodule presented close proximity to the maxillary tuberosity. No swelling or abnormality was observed in the palatal region. Despite being edentulous, the patient did not use a dental prosthesis. Radiographically, a well-delimited, ovoid, and radiopaque lesion was observed on the edentulous alveolar ridge (
<xref ref-type="fig" rid="F3">Fig. 3</xref>
). The main hypothesis of diagnosis was peripheral osteoma. Under local anesthesia, an excisional biopsy was performed and the specimen sent for histopathological analysis. Microscopically, in addition to the features described in case 1, a fibrous capsule around the lesion was noted (
<xref ref-type="fig" rid="F4">Fig. 4</xref>
). The definitive diagnosis of peripheral osteoma, compact type, was established. The patient did not present postoperative complications and has been disease free after 1 year of treatment.</p>
</sec>
</sec>
<sec sec-type="discussion">
<title>Discussion</title>
<p>The pathogenesis of osteomas is unclear. Some investigators considered it a true neoplasm, while others classified it as a developmental anomaly.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B10">10</xref>
,
<xref ref-type="bibr" rid="B18">18</xref>
Osteomas exhibit continuous growth rather than growth cessation at adulthood, and this characteristic is the major feature distinguishing them from other bony exostoses,
<xref ref-type="bibr" rid="B3">3</xref>
,
<xref ref-type="bibr" rid="B5">5</xref>
supporting a neoplastic origin. In addition, the possibility of a reactive mechanism, triggered by trauma or infection has also been suggested.
<xref ref-type="bibr" rid="B5">5</xref>
,
<xref ref-type="bibr" rid="B18">18</xref>
</p>
<p>Mandibular peripheral osteomas occur predominantly in the angle or condyle, followed by the molar area and ramus, as occurred in case 1. Intraoral cases occur frequently in the lingual molar-premolar area.
<xref ref-type="bibr" rid="B19">19</xref>
In contrast, apart from the maxillary sinus and those cases associated with Gardner syndrome, maxillary osteomas are rare.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B3">3</xref>
,
<xref ref-type="bibr" rid="B7">7</xref>
-
<xref ref-type="bibr" rid="B17">17</xref>
Case 2 seemed to be the eighteenth case of maxillary osteoma published in the English-language literature (
<xref ref-type="table" rid="T1">Table 1</xref>
).</p>
<p>Clinically, they appear circumscribed, usually rounded and protuberant,
<xref ref-type="bibr" rid="B3">3</xref>
,
<xref ref-type="bibr" rid="B5">5</xref>
and are characterized by very slow and continuous growth.
<xref ref-type="bibr" rid="B10">10</xref>
Peripheral osteoma presents as a unilateral, sessile, or pedunculated mass ranging from 1 to 4 cm in diameter,
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B4">4</xref>
,
<xref ref-type="bibr" rid="B6">6</xref>
as observed in both cases. Osteomas, usually asymptomatic, often remain undetected unless incidentally found on a routine radiographic survey or until they cause facial asymmetry or functional impairment.
<xref ref-type="bibr" rid="B2">2</xref>
,
<xref ref-type="bibr" rid="B4">4</xref>
However, depending upon the location, occlusal dysfunction, facial asymmetry, headaches, facial pain, and limited mandibular movements may occur,
<xref ref-type="bibr" rid="B2">2</xref>
mainly in condylar osteomas.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B4">4</xref>
Although most peripheral maxillary osteomas (80%) are located on the alveolar process,
<xref ref-type="bibr" rid="B3">3</xref>
,
<xref ref-type="bibr" rid="B13">13</xref>
as observed in case 2, they rarely cause difficulties in mastication.
<xref ref-type="bibr" rid="B3">3</xref>
</p>
<p>Radiographically, the presence of an oval, radiopaque, well-circumscribed mass attached by a broad base or pedicle to the affected cortical bone is a hallmark of peripheral osteomas.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B19">19</xref>
The differential diagnosis includes exostoses and several pathologic processes including inflammatory and neoplastic lesions such as a sclerotic pattern of chronic osteomyelitis, peripheral ossifying fibroma, periosteal osteoblastoma, osteoid osteoma, and parosteal osteosarcoma.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B4">4</xref>
Distinguishing between exostosis and osteoma may be difficult. The clinical features are essential in determining the final diagnosis since both the conditions present similar histopathological features. Exostoses are usually multiple and located in areas of attached gingival mucosa. Solitary exostoses are rare and occur frequently associated with a local trauma or in regions that received gingival or cutaneous graft.
<xref ref-type="bibr" rid="B20">20</xref>
,
<xref ref-type="bibr" rid="B21">21</xref>
In the present cases, none of these features were observed.</p>
<p>On the other hand, central osteomas may cause more difficulty in diagnosis. Fibrous dysplasia, central ossifying fibroma, odontoma, osteoblastoma, chondroma, cementoblastoma, Paget's disease, and central osteosarcoma should be added to the differential diagnosis of central osteomas.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B4">4</xref>
,
<xref ref-type="bibr" rid="B10">10</xref>
Moreover, pain is an important symptom in 30% of central osteomas.
<xref ref-type="bibr" rid="B10">10</xref>
</p>
<p>Histological classification differentiates among compact or ivory (dense, compact bone), cancellous (soft, spongy bone), and mixed-type osteomas.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B5">5</xref>
,
<xref ref-type="bibr" rid="B19">19</xref>
The compact osteoma comprises dense, compact bone with few marrow spaces, as observed in our cases. The cancellous osteoma is characterized by bony trabeculae and fibro-fatty marrow enclosing osteoblasts, with architecture resembling mature bone.
<xref ref-type="bibr" rid="B1">1</xref>
In the jaw, the compact osteoma is the most common histological subtype,
<xref ref-type="bibr" rid="B22">22</xref>
similar to the current cases.</p>
<p>In patients with multiple maxillofacial osteomas, Gardner syndrome should be considered as a possible diagnosis. In addition to multiple osteomas, cutaneous sebaceous cysts, multiple supernumerary teeth, and colorectal polyposis might be also observed.
<xref ref-type="bibr" rid="B1">1</xref>
,
<xref ref-type="bibr" rid="B23">23</xref>
In our cases, the lesions were solitaries, there were no teeth abnormalities, and no intestinal complaints were reported.</p>
<p>The optimal treatment is surgical, recurrence is rare, and there has been no report of malignant transformation.
<xref ref-type="bibr" rid="B1">1</xref>
The postoperative follow-up should include periodic clinical and radiographic studies.</p>
<p>In summary, osteomas occur predominantly in the maxillofacial region, appearing as slow growing and well-circumscribed, lobulated masses. However, maxillary osteomas are extremely rare lesions and located mainly on the alveolar ridge. The ideal treatment is conservative surgical removal and recurrence is uncommon.</p>
</sec>
</body>
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<floats-group>
<fig id="F1" position="float">
<label>Fig. 1</label>
<caption>
<p>A. Computed tomography (CT) scan reveals a well-circumscribed, bone-like, hyperdense image located on the left mandibular angle. B. CT image shows a lesion with a lobulated surface.</p>
</caption>
<graphic xlink:href="isd-42-99-g001"></graphic>
</fig>
<fig id="F2" position="float">
<label>Fig. 2</label>
<caption>
<p>Histopathological examination shows compact and mature bone, with scarce lacunae and marrow spaces filled by a connective tissue (H&E stain, ×100).</p>
</caption>
<graphic xlink:href="isd-42-99-g002"></graphic>
</fig>
<fig id="F3" position="float">
<label>Fig. 3</label>
<caption>
<p>Periapical radiograph shows a well delimited, ovoid, and radiopaque lesion observed on the edentulous alveolar ridge.</p>
</caption>
<graphic xlink:href="isd-42-99-g003"></graphic>
</fig>
<fig id="F4" position="float">
<label>Fig. 4</label>
<caption>
<p>Histopathological examination shows maxillary osteoma, compact type, circumscribed by a fibrous capsule around the lesion (H&E stain, ×100).</p>
</caption>
<graphic xlink:href="isd-42-99-g004"></graphic>
</fig>
<table-wrap id="T1" position="float">
<label>Table 1</label>
<caption>
<p>Summary of the clinical and radiological features of maxillary osteomas previously published in the English-language literature</p>
</caption>
<graphic xlink:href="isd-42-99-i001"></graphic>
<table-wrap-foot>
<fn>
<p>
<sup>*</sup>
NA: Not available,
<sup>**</sup>
EA: Edentulous area,
<sup></sup>
RP: Radiopaque, WD: well-defined, ID: ill-defined</p>
</fn>
</table-wrap-foot>
</table-wrap>
</floats-group>
</pmc>
</record>

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