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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Co-Occurence of Florid Cemento-Osseous Dysplasia and Simple Bone Cyst: a Case Report</title><author><name sortKey="Rao, Kumuda Arvind" sort="Rao, Kumuda Arvind" uniqKey="Rao K" first="Kumuda Arvind" last="Rao">Kumuda Arvind Rao</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author><author><name sortKey="Shetty, Shishir Ram" sort="Shetty, Shishir Ram" uniqKey="Shetty S" first="Shishir Ram" last="Shetty">Shishir Ram Shetty</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author><author><name sortKey="Babu, Subhas G" sort="Babu, Subhas G" uniqKey="Babu S" first="Subhas G." last="Babu">Subhas G. Babu</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author><author><name sortKey="Castelino, Renita Lorina" sort="Castelino, Renita Lorina" uniqKey="Castelino R" first="Renita Lorina" last="Castelino">Renita Lorina Castelino</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">24421997</idno><idno type="pmc">3886077</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3886077</idno><idno type="RBID">PMC:3886077</idno><idno type="doi">10.5037/jomr.2011.2305</idno><date when="2011">2011</date><idno type="wicri:Area/Pmc/Corpus">001E91</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">001E91</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Co-Occurence of Florid Cemento-Osseous Dysplasia and Simple Bone Cyst: a Case Report</title><author><name sortKey="Rao, Kumuda Arvind" sort="Rao, Kumuda Arvind" uniqKey="Rao K" first="Kumuda Arvind" last="Rao">Kumuda Arvind Rao</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author><author><name sortKey="Shetty, Shishir Ram" sort="Shetty, Shishir Ram" uniqKey="Shetty S" first="Shishir Ram" last="Shetty">Shishir Ram Shetty</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author><author><name sortKey="Babu, Subhas G" sort="Babu, Subhas G" uniqKey="Babu S" first="Subhas G." last="Babu">Subhas G. Babu</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author><author><name sortKey="Castelino, Renita Lorina" sort="Castelino, Renita Lorina" uniqKey="Castelino R" first="Renita Lorina" last="Castelino">Renita Lorina Castelino</name><affiliation><nlm:aff id="aff1"><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></nlm:aff></affiliation></author></analytic><series><title level="j">Journal of Oral & Maxillofacial Research</title><idno type="eISSN">2029-283X</idno><imprint><date when="2011">2011</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><title>ABSTRACT</title><sec sec-type="background"><title>Background</title><p>The purpose of this report is to present a rare case of co-occurrence of
florid cemento-osseous dysplasia with simple bone cyst in a middle aged
Asian woman. Most of the reported cases are isolated cases of simple bone
cyst or florid cemento-osseous dysplasia, but co-occurrence of these two
entities is extremely rare.</p></sec><sec sec-type="methods"><title>Methods</title><p>The authors report a 41 year old female patient with co-occurrence of
mandibular florid cemento-osseous dysplasia with simple bone cyst. A
thorough clinical and radiological examination was carried out.</p></sec><sec sec-type="results"><title>Results</title><p>It was diagnosed mandibular cyst with possible co-occurrence of florid
cemento-osseous dysplasia. Surgical exploration of the multilocular lesion
was applied. Since, the patient was symptomatic at the time of presentation
utmost caution was taken during the surgical procedure as florid
cemento-osseous dysplasia is associated with hypo-vascularity of the
affected bone. Based on histopathological, as well as supporting
clinico-radiological findings a confirmative diagnosis of florid
cemento-osseous dysplasia co-occurring with simple bone cyst was made.
Patient was followed-up for a period of six months and was reported to be
asymptomatic.</p></sec><sec sec-type="conclusions"><title>Conclusions</title><p>Timely diagnosis and well planned treatment is important to obtain a good
prognosis when a rare co-occurrence of two or more bone lesions affects the
jaws.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Ong, St" uniqKey="Ong S">ST Ong</name></author><author><name sortKey="Siar, Ch" uniqKey="Siar C">CH Siar</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Pitak Arnnop, P" uniqKey="Pitak Arnnop P">P Pitak-Arnnop</name></author><author><name sortKey="Dhanuthai, K" uniqKey="Dhanuthai K">K Dhanuthai</name></author><author><name sortKey="Chaine, A" uniqKey="Chaine A">A Chaine</name></author><author><name sortKey="Bertrand, Jc" uniqKey="Bertrand J">JC Bertrand</name></author><author><name sortKey="Bertolus, C" uniqKey="Bertolus C">C Bertolus</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gunduz, K" uniqKey="Gunduz K">K Gündüz</name></author><author><name sortKey="Avsever, H" uniqKey="Avsever H">H Avsever</name></author><author><name sortKey="Karacayli, U" uniqKey="Karacayli U">U 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<front><journal-meta><journal-id journal-id-type="nlm-ta">J Oral Maxillofac Res</journal-id><journal-id journal-id-type="iso-abbrev">J Oral Maxillofac Res</journal-id><journal-id journal-id-type="publisher-id">JORM</journal-id><journal-title-group><journal-title>Journal of Oral & Maxillofacial Research</journal-title></journal-title-group><issn pub-type="epub">2029-283X</issn><publisher><publisher-name>Stilus Optimus</publisher-name><publisher-loc>Kaunas, Lithuania</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">24421997</article-id><article-id pub-id-type="pmc">3886077</article-id><article-id pub-id-type="publisher-id">v2n3e5ht</article-id><article-id pub-id-type="doi">10.5037/jomr.2011.2305</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Co-Occurence of Florid Cemento-Osseous Dysplasia and Simple Bone Cyst: a Case Report</article-title></title-group><contrib-group><contrib id="contrib1" contrib-type="author" corresp="yes"><name><surname>Rao</surname><given-names>Kumuda Arvind</given-names></name><xref ref-type="aff" rid="aff1">1</xref></contrib><contrib id="contrib2" contrib-type="author"><name><surname>Shetty</surname><given-names>Shishir Ram</given-names></name><xref ref-type="aff" rid="aff1">1</xref></contrib><contrib id="contrib3" contrib-type="author"><name><surname>Babu</surname><given-names>Subhas G.</given-names></name><xref ref-type="aff" rid="aff1">1</xref></contrib><contrib id="contrib4" contrib-type="author"><name><surname>Castelino</surname><given-names>Renita Lorina</given-names></name><xref ref-type="aff" rid="aff1">1</xref></contrib></contrib-group><aff id="aff1" rid="aff1"><sup>1</sup><institution>Department of Oral and Maxillofacial Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University</institution><addr-line>Deralakatte, Mangalore, Karnataka</addr-line><country>India.</country></aff><author-notes><corresp>Kumuda Arvind Rao,
<institution>Department of Oral Medicine and Radiology, AB Shetty Memorial
Institute of Dental Sciences.</institution><addr-line>Nitte University, Deralakatte, Mangalore 575018,
Karnataka</addr-line><country>India</country>Phone: +91 944 8696705<email>kumuda_arht@rediffmail.com</email></corresp></author-notes><pub-date pub-type="collection"><season>Jul-Sep</season><year>2011</year></pub-date><pub-date pub-type="epub"><day>1</day><month>10</month><year>2011</year></pub-date><volume>2</volume><issue>3</issue><elocation-id>e5</elocation-id><history><date date-type="received"><day>18</day><month>3</month><year>2011</year></date><date date-type="accepted"><day>14</day><month>4</month><year>2011</year></date></history><permissions><copyright-statement> Copyright © Rao KA, Shetty SR, Babu SG, Castelino RL.
Published in the JOURNAL OF ORAL & MAXILLOFACIAL RESEARCH
(http://www.ejomr.org), 1 October 2011.</copyright-statement><copyright-year>2011</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-nd/3.0/"><license-p>This is an open-access article, first published in the JOURNAL OF
ORAL & MAXILLOFACIAL RESEARCH, distributed under the terms of the
Creative Commons Attribution-Noncommercial-No Derivative Works 3.0 Unported
License (<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by-nc-nd/3.0/">http://creativecommons.org/licenses/by-nc-nd/3.0/</ext-link>), which permits unrestricted non-commercial use, distribution, and
reproduction in any medium, provided the original work and is properly
cited. The copyright, license information and link to the original
publication on <ext-link ext-link-type="uri" xlink:href="http://www.ejomr.org">http://www.ejomr.org</ext-link> must be included.</license-p></license></permissions><self-uri xlink:type="simple" xlink:href="http://www.ejomr.org/JOMR/archives/2011/3/e5/v2n3e5ht.htm"></self-uri><abstract><title>ABSTRACT</title><sec sec-type="background"><title>Background</title><p>The purpose of this report is to present a rare case of co-occurrence of
florid cemento-osseous dysplasia with simple bone cyst in a middle aged
Asian woman. Most of the reported cases are isolated cases of simple bone
cyst or florid cemento-osseous dysplasia, but co-occurrence of these two
entities is extremely rare.</p></sec><sec sec-type="methods"><title>Methods</title><p>The authors report a 41 year old female patient with co-occurrence of
mandibular florid cemento-osseous dysplasia with simple bone cyst. A
thorough clinical and radiological examination was carried out.</p></sec><sec sec-type="results"><title>Results</title><p>It was diagnosed mandibular cyst with possible co-occurrence of florid
cemento-osseous dysplasia. Surgical exploration of the multilocular lesion
was applied. Since, the patient was symptomatic at the time of presentation
utmost caution was taken during the surgical procedure as florid
cemento-osseous dysplasia is associated with hypo-vascularity of the
affected bone. Based on histopathological, as well as supporting
clinico-radiological findings a confirmative diagnosis of florid
cemento-osseous dysplasia co-occurring with simple bone cyst was made.
Patient was followed-up for a period of six months and was reported to be
asymptomatic.</p></sec><sec sec-type="conclusions"><title>Conclusions</title><p>Timely diagnosis and well planned treatment is important to obtain a good
prognosis when a rare co-occurrence of two or more bone lesions affects the
jaws.</p></sec></abstract><kwd-group><kwd>florid cemento-osseous dysplasia</kwd><kwd>mandibular diseases</kwd><kwd>mandibular neoplasms</kwd><kwd>surgery</kwd><kwd>oralflorid cemento-osseous dysplasia</kwd><kwd>mandibular diseases</kwd><kwd>mandibular neoplasms</kwd><kwd>oral surgery.</kwd></kwd-group></article-meta></front><body><sec><title>INTRODUCTION</title><p>Florid cemento-osseous dysplasia (FCOD) refers to a group of fibro-osseous lesions,
which are exuberant, multiquadrant and arise from the tooth-bearing area of the jaws
[<xref ref-type="bibr" rid="B1">1</xref>]. FCOD is a benign jaw lesion and is
discovered most frequently in the mandible of middle-aged black female [<xref ref-type="bibr" rid="B2">2</xref>]. It usually presents a multiple radiopaque
bone/cemetum-like masses distributed throughout the jaw [<xref ref-type="bibr" rid="B3">3</xref>].</p><p>FCOD lesions have a striking tendency towards bilateral, often quite symmetrical,
location, and it is not unusual to find extensive lesions in all 4 posterior
(molar-premolar region) quadrants of the jaw [<xref ref-type="bibr" rid="B4">4</xref>]. The word "florid" was introduced to describe the wide
spread, extensive manifestations of the disease in multiple quadrants of the jaws
[<xref ref-type="bibr" rid="B5">5</xref>]. The various synonyms used are
multiple enostoses, multiple cemento-ossifying fibromas, multiple periapical
osteofibromatosis, florid cement-osseous dysplasia and gigantiform cementom [<xref ref-type="bibr" rid="B2">2</xref>]. In 2005, World Health Organization
subdivided Cemento-Osseous Dysplasias (CODs) into periapical, florid and other CODs
[<xref ref-type="bibr" rid="B6">6</xref>]. The definite diagnosis of these 3
diseases cannot be reached by clinical ground, but only by histopathologic
examination [<xref ref-type="bibr" rid="B2">2</xref>]. Although, the disease may be
totally asymptomatic, some patients present with pain, swelling, purulent discharge
and sequestrum formation [<xref ref-type="bibr" rid="B7">7</xref>]. Rare reports of
its association with simple bone cysts (SBC) are present usually when the cases
reported are symptomatic. Melrose and co-workers [<xref ref-type="bibr" rid="B8">8</xref>] were the first to observe this association in their series of 34
cases, where 14 patients had concurrent, biopsy-proven simple bone cyst. FCOD is not
associated with any other extragnathic abnormalities and there are no abnormalities
in blood chemistry of patients [<xref ref-type="bibr" rid="B5">5</xref>].</p><p>Most benign fibro-osseous lesions of jaws are asymptomatic and slowly progressing
[<xref ref-type="bibr" rid="B9">9</xref>]. Those benign fibro-osseous lesions
that present as an atypical radiographic appearance require a detailed clinical,
radiographic and laboratory workup to arrive at a diagnosis [<xref ref-type="bibr" rid="B10">10</xref>]. For the asymptomatic patient, the best management consists
of regular recall examinations with prophylaxis and reinforcement of good home
hygiene care to control periodontal disease and prevent tooth loss [<xref ref-type="bibr" rid="B11">11</xref>].</p><p>The case presented here highlights a rare combination of FCOD co-existing with a
multilocular radiolucency. Computed tomography (CT), because of its ability to give
three-dimensional axial, sagittal, and frontal views, is useful in the evaluation of
these lesions. This paper presents the case of a patient, who was diagnosed with
FCOD on the basis of clinical and radiographic findings. SBC was diagnosed on the
basis of surgical and histopathological findings. We hereby report one rare case of
co-occurrence of florid cemento-osseous dysplasia with simple bone cyst presenting
with symptoms of pain and swelling in the jaw.</p></sec><sec><title>CASE REPORT</title><p>41 year old female patient reported to the Department of Oral and Maxillofacial
Radiology, AB Shetty Memorial Institute of Dental Sciences, Nitte University,
Deralakatte, Mangalore, Karnataka, India, with a complaint of pain in the right
posterior region of the mandible since 4 days. The pain radiated to the right side
of the face, and was continuous in nature. Patient gave a history of extraction of a
tooth in the same region 2 years ago. Tooth was extracted due to decay, after which
timely healing did not occur and was associated with mild intermittent pain. Patient
also complained of an enlargement of the jaw in the same site. No associated
systemic symptoms were reported by the patient. Patient's medical history was
non-contributory.</p><p>Extraoral examination revealed single diffuse smooth swelling on the right side of
the jaw, approximately 1 x 1 cm in size, with normal overlying skin (<xref ref-type="fig" rid="fig1">Figure 1</xref>). The swelling was hard in consistency
and tender on palpation. Right submandibular lymph nodes were tender on
palpation.</p><fig id="fig1" orientation="portrait" position="float"><label>Figure 1</label><caption><p>Clinical photograph of the patient showing swelling on the right side of the
cheek (white arrow).</p></caption><graphic xlink:href="jomr-02-e5-g001"></graphic></fig><p>Intraoral examination showed an unhealed extraction socket seen in relation to #48.
Surface was covered by slough and debris. Surrounding mucosa appeared to be normal
in colour. No discharge was noticed (<xref ref-type="fig" rid="fig2">Figure
2</xref>).</p><fig id="fig2" orientation="portrait" position="float"><label>Figure 2</label><caption><p>Intraoral photograph showing unhealed extraction socket (encircled in
black).</p></caption><graphic xlink:href="jomr-02-e5-g002"></graphic></fig><p>Palpation revealed swelling with bony hard consistency and soft tender. Buccolingual
cortical expansion was experienced. The area surrounding the socket was tender.
Normal response was seen in all the teeth, when electric pulp testing was carried
out.</p><p>An intraoral periapical view of the area revealed a well defined radiolucency.
Superiorly, it extended 0.5 cm from the crest of the edentulous alveolar ridge and
presented with scalloped border, with incomplete septa running into the radiolucency
(<xref ref-type="fig" rid="fig3">Figure 3</xref>). The inferior and anterior
extent of the radiolucency were not clear, hence an OPG was suggested.</p><fig id="fig3" orientation="portrait" position="float"><label>Figure 3</label><caption><p>Intraoral periapical radiograph showing well defined radiolucency with
scalloped border (white arrows).</p></caption><graphic xlink:href="jomr-02-e5-g003"></graphic></fig><p>Orthopantomography revealed well defined radiolucency in the body of mandible on the
right side, measuring approximately 3 x 4 cm. It was present in the region of teeth
#46, #47 and #48 involving the periapical area of #45 anteriorly, up to the anterior
border of the ramus posteriorly. The radiolucent area extended superiorly up to 0.5
cm from the edentulous alveolar crest and 0.5 cm above the lower border of the
mandible. The course of the mandibular canal could not be traced. Multiple mixed
radio-opacities were present around the periapical region of all mandibular teeth
above the level of the mandibular canal. The radiolucency was also observed at the
periapices of teeth #18, #12, #26 and #28 in the maxilla suggestive of a
fibro-osseous lesion. The radio opacities presented with a radiolucent halo around
them (<xref ref-type="fig" rid="fig4">Figure 4</xref>). Mandibular occlusal view of
the same region revealed expansion of the buccal and lingual cortical plates with
presence of multiple internal loculations (<xref ref-type="fig" rid="fig5">Figure
5</xref>). Serum work-up revealed no abnormalities.</p><fig id="fig4" orientation="portrait" position="float"><label>Figure 4</label><caption><p>Orthopantamogram showing multilocular radiolucency on the right body of the
mandible (white arrow) and multiple radio opacities in all the quadrants of
both jaws (yellow arrows).</p></caption><graphic xlink:href="jomr-02-e5-g004"></graphic></fig><fig id="fig5" orientation="portrait" position="float"><label>Figure 5</label><caption><p>Mandibular occlusal radiograph showing cortical expansion and internal
loculations (encircled in yellow).</p></caption><graphic xlink:href="jomr-02-e5-g005"></graphic></fig><p>Based on these radiological findings, lists of differential diagnoses for the
radiolucenct, as well as radio-opaque areas were made. For the multilocular
radiolucency ameloblastoma, odontogenic keratocyst, simple bone cysts were enlisted
and for the diffuse radioopaque lesion, diffuse sclerosing osteomyelitis, florid
cemento-osseous dysplasia, Paget's disease were enlisted as differential
diagnoses.</p><p>To determine the boundaries of the lesion preoperatively, a multidetector CT scan was
made. It revealed, a well defined expansile osteolytic lesion measuring
approximately 1.7 x 2.3 x 2.7 cm in size in the right body of the mandible at the
level of the molar teeth. There is a narrow zone of transition (<xref ref-type="fig" rid="fig6">Figure 6</xref>).</p><fig id="fig6" orientation="portrait" position="float"><label>Figure 6</label><caption><p>CT section showing a well defined expansile osteolytic lesion in the right
body of the mandible (yellow arrow).</p></caption><graphic xlink:href="jomr-02-e5-g006"></graphic></fig><p>Multiple patchy well defined hyperdense lesions with hypodense rim were observed in
the body of mandible located above the level of mandibular canal, between the
inter-radicular and inter-dental areas of maxilla and mandible (<xref ref-type="fig" rid="fig7">Figure 7</xref> and <xref ref-type="fig" rid="fig8">Figure 8</xref>).
The CT scan findings were consistent with florid cemento-osseous dysplasia - stage
III (mature stage).</p><fig id="fig7" orientation="portrait" position="float"><label>Figure 7</label><caption><p>Coronal CT section showing multiple patchy well defined hyperdense lesions
with hypodense rim were observed above the level of mandibular canal (yellow
arrow).</p></caption><graphic xlink:href="jomr-02-e5-g007"></graphic></fig><fig id="fig8" orientation="portrait" position="float"><label>Figure 8</label><caption><p>Sagittal CT section showing multiple patchy well defined hyperdense lesions
with hypodense rim (yellow arrows).</p></caption><graphic xlink:href="jomr-02-e5-g008"></graphic></fig><p>Surgical exploration of the multilocular lesion showed connective tissue that was
tightly adherent to the surrounding bone and was devoid of epithelium. The area was
thoroughly cleaned with curette and the adjacent bony areas were sent for
histopathological examination. Internal titanium plate fixation was done to the
lower border of the mandible to avoid pathologic fracture due to thinning of the
cortex (<xref ref-type="fig" rid="fig9">Figure 9</xref>).</p><fig id="fig9" orientation="portrait" position="float"><label>Figure 9</label><caption><p>Postoperative orthopantamogram showing internal plate fixation (yellow
arrows).</p></caption><graphic xlink:href="jomr-02-e5-g009"></graphic></fig><p>Histological examination of the tissue lining the bony cavity revealed presence of
fibrocellular connective tissue with osteoid areas as well as mineral deposition
within them. The connective tissue shows spindle shaped fibroblasts and collagen
fibres. Blood vessels and chronic inflammatory cells in the form of lymphocytes were
additionally identified (<xref ref-type="fig" rid="fig10">Figure 10</xref>).</p><fig id="fig10" orientation="portrait" position="float"><label>Figure 10</label><caption><p>Hematoxylin and eosin stained histopathological section (original
magnification x10) of periapical bone and buccal cortex in the region of
teeth #45 and #46 showing immature bone with osteocytes.</p></caption><graphic xlink:href="jomr-02-e5-g010"></graphic></fig><p>Histopathological examination of stained slides of periapical bone and buccal cortex
in the region of teeth #45 and #46 shows immature bone with osteocytes. Numerous
capillaries and resting lines are also seen (<xref ref-type="fig" rid="fig11">Figure
11</xref>).</p><fig id="fig11" orientation="portrait" position="float"><label>Figure 11</label><caption><p>Hematoxylin and eosin stained histopathological section (original
magnification x40) of cavity lining showing fibrocellular connective tissue
with osteiod areas within them (black arrow).</p></caption><graphic xlink:href="jomr-02-e5-g011"></graphic></fig><p>Based on these histopathological, as well as supporting clinico-radiological findings
a confirmative diagnosis of florid cemento-osseous dysplasia co-occurring with
simple bone cyst was made.</p></sec><sec sec-type="discussion"><title>DISCUSSION</title><p>FCOD is the most common pathologic condition of the jaws that occurs as radiopacities
in multiple quadrants of the tooth-bearing regions of the jaws [<xref ref-type="bibr" rid="B12">12</xref>]. This disorder is strictly localized to the
tooth bearing areas and not associated with any other skeletal disease [<xref ref-type="bibr" rid="B13">13</xref>]. However, the etiopathogenesis is not
clear. Waldron et al. [<xref ref-type="bibr" rid="B13">13</xref>] have proposed that
reactive or dysplastic changes in the periodontal ligament might be a cause for the
disease. This condition has also been classified by various authors as sclerosing
osteomyelitis, sclerosing osteitis, sclerotic cemental masses, gigantiform
cementoma, and various other terms [<xref ref-type="bibr" rid="B5">5</xref>].</p><p>FCOD is a benign fibro-osseous lesion in which mature bone is replaced with a woven
bone in a matrix of fibrous connective tissue [<xref ref-type="bibr" rid="B14">14</xref>]. Similar histopathological features were noted in our case.</p><p>FCOD is a rare disease entity especially in the Indian population; only 2% cases have
been reported among the Indians in the literature [<xref ref-type="bibr" rid="B13">13</xref>]. These lesions are most commonly seen in middle-aged black women,
although it may also occur in Caucasians and Asians [<xref ref-type="bibr" rid="B3">3</xref>]. Our patient was a 41 year old woman of Asian origin.</p><p>Clinically, these lesions are often asymptomatic and may present as an incidental
radiological findings [<xref ref-type="bibr" rid="B15">15</xref>]. FCOD occasionally
is expansile, and patients may report experiencing pain [<xref ref-type="bibr" rid="B14">14</xref>]. Similar complaints were reported by our patient. Symptoms
such as dull pain or drainage are almost always associated with exposure of
sclerotic calcified masses in the oral cavity. No such features were seen in our
case. This may occur as a result of progressive alveolar atrophy under a denture or
after extraction of teeth in the affected area [<xref ref-type="bibr" rid="B16">16</xref>]. The patient presented herein, underwent tooth extraction 2 years
ago after which the healing did not occur. Occasionally patients may also present
with complaint of intermittent, poorly localized pain in the affected bone,
especially when a simple bone cyst has developed within the lesion. Extensive
lesions often have an associated bone deformation [<xref ref-type="bibr" rid="B17">17</xref>]. Progressive increase in the bulk of the lesion was seen in case
report presented by Miyake and Nagahata [<xref ref-type="bibr" rid="B18">18</xref>].
Similar history was presented by our patient. Waldron suggested that periapical
cemento-osseous dysplasia and focal cemento-osseous dysplasia may also develop into
florid cemento-osseous dysplasia [<xref ref-type="bibr" rid="B19">19</xref>,<xref ref-type="bibr" rid="B20">20</xref>].</p><p>A simple bone cyst can be found in association with benign fibro-osseous lesions such
as cemento-osseous dysplasia and fibrous dysplasia [<xref ref-type="bibr" rid="B15">15</xref>]. Some reports have described an association between solitary bone
cysts and fibro-osseous lesions including fibrous dysplasia and cemento-osseous
dysplasias [<xref ref-type="bibr" rid="B21">21</xref>]. FCOD with concomitant simple
bone cysts is not common [<xref ref-type="bibr" rid="B22">22</xref>]. The
pathogenesis of simple bone cysts is largely known. Venous obstruction and blockage
of interstitial fluid drainage, in these areas of rapidly growing and remodelling
cancellous bone, may lead to formation of the simple bone cysts [<xref ref-type="bibr" rid="B15">15</xref>].</p><p>In our case, the diagnosis of FCOD was made clinico-radiologically and
histopathologically. An elective surgical procedure including biopsy was not
performed, because of the risk of secondary infection. Secondary infection occurs in
such lesions due to abundant cementum formation and poor vascularity [<xref ref-type="bibr" rid="B17">17</xref>].</p><p>Radiographically, FCOD is characterized by extensive sclerotic areas, often involving
the posterior quadrants of the mandible and maxilla in a symmetric fashion [<xref ref-type="bibr" rid="B23">23</xref>]. FCOD is a diffuse form characterised by
multiple periapical lesions involving one or both the jaws. It occurs around the
root apices of vital tooth in middle-aged women with a predilection for mandibular
incisors. In the early stage, it appears as a well defined radiolucent lesion, which
gradually becomes totally radiopaque with a thin lucent rim in the mature stages
[<xref ref-type="bibr" rid="B24">24</xref>].</p><p>Asymptomatic lesions do not require intervention because, complete resection of the
lesion would be impractical as it usually occupies most of the mandible and maxilla
[<xref ref-type="bibr" rid="B16">16</xref>]. When surgical intervention is
indicated, a remodelling resection is recommended for aesthetic reasons [<xref ref-type="bibr" rid="B25">25</xref>]. Histopathologically, FCOD is composed of a
proliferating fibrous connective tissue stroma containing foci of cementum along
with the presence of osteoid or bone. More advanced lesions show an increase in
mineralization. In FCOD, large sclerotic masses are formed that are hypocellular and
extremely dense with small marrow spaces and few Haversian systems [<xref ref-type="bibr" rid="B21">21</xref>].</p><p>The simple bone cysts that occur without any association with cemento-osseous
dysplasia tend to heal better after surgery than those associated with
cemento-osseous dysplasia [<xref ref-type="bibr" rid="B26">26</xref>]. The
histological features of simple bone cysts are mostly non-specific, and when a
tissue is submitted from this lesion, a microscopic examination will reveal only a
strip of fibrous connective tissue, occasionally with an associated rim of bone
[<xref ref-type="bibr" rid="B15">15</xref>].</p><p>Expansion of the buccal and lingual cortical plates is not observed in FCOD unless
associated with cystic changes [<xref ref-type="bibr" rid="B27">27</xref>]. A
similar finding supported our case.</p><p>As differential diagnosis we can include Paget's disease which would present
radiologically with cotton-wool appearance and diffuse sclerosing osteomyelitis
which can also be a complication associated with FCOD. Paget's disease is also
characterized by deformities of multiple bones and produces biochemical serum
changes, such as elevated alkaline phosphate levels [<xref ref-type="bibr" rid="B15">15</xref>]. No such biochemical alterations or other bone deformities were
present in present case.</p><p>Regular follow-up during six month was maintained and patient reported to be
asymptomatic after the surgery.</p></sec><sec sec-type="conclusions"><title>CONCLUSIONS</title><p>The management of florid cemento-osseous dysplasia may not be satisfactory, since the
disease process may run for a very long time without any symptoms. When patients are
asymptomatic, optimum oral hygiene has to be maintained to avoid tooth loss and
periodontal disease. Elective intraoral procedures have to be avoided due to the
associated risk of infection or subsequent osteomyelitis and fracture. When the
patient is symptomatic secondary to a tooth pain, the tooth may be managed
endodontically by avoiding extractions. Cases with secondary predisposed factor of
infection are difficult and complicated to manage. Since our patient will require
frequent scans during the follow-ups, imaging modalities with lesser radiation doses
have to be employed. Surgery, when required should be carried out with a minimal
invasion. The treatment options should be easily available and accessible,
affordable to the masses without hampering the aesthetics and function of the
patient.</p></sec></body><back><ack><sec sec-type="acknowledgments and disclosure statements"><title>ACKNOWLEDGMENTS AND DISCLOSURE STATEMENTS</title><p>The authors declare that they have no conflict of interests. We acknowledge Dr.
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