PCOD
PCOD
PCOD
Objectives. The objective of this study was to identify treatment indications for symptomatic and asymptomatic florid cemento-
osseous dysplasia (FCOD) in adult patients and explore relationships between clinical variables and radiographic findings (PROS-
PERO # CRD42023411228).
Study Design. A systematic review was conducted by independent investigators using databases: PubMed, ProQuest, Embase,
Web of Science, Dentistry and Oral Sciences Database (DOSS), and TRIP to identify studies on FCOD treatment options in adults.
Inclusion criteria for this systematic review included: originally in English; open-access; published between 2001 and 2021. Pre-
ferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines and the Joanna Briggs Institute (JBI) Critical
Appraisal checklist were used for reporting and quality assessment of each study.
Results. From the initial 122 studies, 11 fit criteria for this systematic review. Eight studies reported symptoms as clinical presenta-
tion of FCOD, 6 reported swelling, and 3 reported infection. Five studies recommended surgical treatment in symptomatic
patients with anatomic structure changes around the lesions, such as presence of necrotic bone and secondary infection. Treat-
ment of asymptomatic FCOD was contraindicated in 10 studies. Dental prophylaxis was recommended.
Conclusion. Dental prophylaxis and monitoring were the most common management strategies for asymptomatic and symptom-
atic FCOD. Surgical curettage with stimulation of bleeding, or pulp vitality testing of specific areas with periapical inflammation
were treatment options for symptomatic cases. <END ABSTRACT> (Oral Surg Oral Med Oral Pathol Oral Radiol YEAR;VOL:
page range) (Oral Surg Oral Med Oral Pathol Oral Radiol 2024;000:1 16)
Osseous dysplasia (OD) is a condition that occurs examinations.4 This systematic review will be concen-
from proliferation of the periodontal ligament fibro- trating on the FCOD subtype.
blasts with bone deposits, cementum, and fibrous tissue For accurate diagnosis of FCOD, periapical and pan-
in the jaw areas of the apex regions of teeth.1 oramic radiographs, and cone-beam computed tomog-
Cemento-osseous dysplasia (COD) are fibro-osseous raphy (CBCT) scans are used.5 In radiography, early
lesions which contain fibrous connective tissue, woven FCOD usually presents as radiolucencies. In later
bone, and cementum as a replacement of normal stages, FCOD presents as mixed radiolucent and radi-
bone.1 COD tends to manifest in 40 to 60-year-old opaque entity.1 In final stages, masses become
Black and Asian women, often presenting as asymp- completely radiopaque. Densely radiopaque lesions
tomatic.2 There are 3 subtypes of COD: focal, periapi- have poor vascularization of surrounding tissue.6
cal, and florid.3 Florid cemento-osseous dysplasia FCOD areas are observed as separated from the roots
(FCOD) is also commonly used interchangeably with of teeth by fibrous tissue and can be associated with
florid osseous dysplasia (FOD). FCOD by definition is simple bone cysts (SBCs).1 When diagnosing COD, it
multifocal COD, present in the tooth bearing areas of is important to use clinical presentation to not misdiag-
either or both jaws. The presence of FCOD is mostly nose as osteosarcomas or ossifying fibromas due to
found as an incidental finding during radiographic their similar histology.1 Although FCOD usually
presents asymptomatically, it can become symptomatic
spontaneously, or if traumatized, biopsied, or having
a
Department of Oral Health Sciences, Temple University Maurice H. tooth extraction / surgical treatment in affected areas
Kornberg School of Dentistry, Philadelphia, PA, USA.
b
due to introduction of bacteria into the bone.7 Conse-
Department of Prosthodontics, Temple University Maurice H. Korn-
berg School of Dentistry, Philadelphia, PA, USA.
quently, biopsy and other surgical interventions in
c
Department of Oral Pathology, Temple University Maurice H.
Kornberg School of Dentistry, Philadelphia, PA, USA.
d
Department of Oral Radiology, Temple University Maurice H. Statement of Clinical Relevance
Kornberg School of Dentistry, Philadelphia, PA, USA.
Corresponding author: Miriam Gabay. E-mail address: miriam. There is no definitive recommendation in literature
gabay@temple.edu
Received for publication Feb 12, 2024; returned for revision May 31,
on treatment of symptomatic or asymptomatic
2024; accepted for publication Jun 13, 2024. FCOD. Some literature states symptomatic patients
Ó 2024 Elsevier Inc. All rights are reserved, including those for text should receive antibiotic therapy, others call for sur-
and data mining, AI training, and similar technologies. gical intervention. This systematic review aimed to
2212-4403/$-see front matter create consensus of optimal management for FCOD.
https://doi.org/10.1016/j.oooo.2024.06.011
1
ARTICLE IN PRESS
ORAL AND MAXILLOFACIAL SURGERY OOOO
2 Gabay et al. && 2024
regions of COD and FCOD are often considered con- applicable articles for the systematic review to be per-
traindicated. formed. The MeSH terms used in this systematic
FCOD is therefore typically diagnosed based on review are (florid cemento-osseous dysplasia) OR
clinical and radiographic presentation. Asymptomatic (florid osseous dysplasia) OR (symptomatic) OR
patients are managed only by being monitored by clini- (asymptomatic) AND (treatment). The filter was set to
cians clinically and radiographically at some regular include cohort, cross-sectional, longitudinal, and retro-
interval, for example, every 6 months.4 For this system- spective studies as there were no randomized clinical
atic review, symptomatic patients were defined as hav- control trials for this subject. The ISSG filter was used
ing swelling, presence of infection, or pain in FCOD for the search. All literature was exported into Zotero
affected areas. 5.0.96.3 reference management software for the man-
Evidence suggests that if secondary infection of agement of literature data (Table I).
FCOD occurs (e.g., complications of biopsy, tooth
extractions, periodontal surgery, etc.) or if the patient Eligibility / selection criteria
becomes symptomatic, surgical treatment for FCOD Inclusion criteria for this systematic review were: all
can be offered.2 studies included must be originally in English; can
There is currently no clear consensus or definitive be from around the world; open-access; published
recommendation in the literature on treatment of symp- between the time frame 2001 and 2021; include adults,
tomatic FCOD. For example, some literature states that ages 19 years or above. There must have been a statisti-
symptomatic patients with infection are to receive anti- cal or descriptive analysis included in these studies. All
biotic therapy as primary treatment,8 whereas other lit- literature must have reported the specific subtype of
erature calls for surgical intervention.2 Similarly, there COD: FCOD. Radiographic imaging must have been
is no definitive recommendation for the management evaluated as a source for diagnosis in all studies being
of (including monitoring regimen) of asymptomatic used. Common treatment outcomes must be evaluated
FCOD. Hence, there is a gap in existing literature post-diagnosis in all studies being used. Outcomes to
regarding recommendations for treatment options in be evaluated included post-surgical recurrence of
symptomatic and asymptomatic FCOD. infections, efficacy of antibiotic therapy on the initial
presence of infection, post-operatory complications,
OBJECTIVES and factors that can contribute to possible complica-
The objectives of this systematic review were to: tions (e.g., diabetes and smoking). Exclusion criteria
included case reports; articles published prior to 2001;
1. Ascertain the treatment indications for patients with and lack of FCOD subtype reported.
FCOD in symptomatic vs asymptomatic adult
patients. Screening criteria
2. Explore the relationships among clinical variables, There was a total of 122 studies found before screening
demographics, and radiographic findings in patients duplicates (Figure 1). There were 40 duplicates found
with symptomatic and asymptomatic FCOD. throughout the databases. Following this, 11 studies
were removed by their title. Seventy-one studies were
assessed for eligibility. Fifty-nine studies were
excluded due to being case reports, being excerpts
METHODS from journals, lack of treatment options, and by
This study is a systematic review conducted by 3 inde- abstract. One study from Embase was not available for
pendent investigators (authors M.G., C.O., and L.D.). retrieval. The remaining 11 studies fulfilled all inclu-
This systematic review was registered in Prospero sion criteria and were therefore included in this system-
(#CRD42023411228). atic review.
for inclusion was clearly defined, study subjects and bias due to the nature of the cross-sectional studies
setting were described in detail, sample size adequate, included and lack of randomization.
objective criteria used for measurement of condition,
whether the condition was measured in a standard and
reliably way for all participants, valid methods used for RESULTS
identification of the condition, were confounding fac- Table III presents the summary of characteristics of the
tors identified, and whether there was an appropriate selected 11 studies. The characteristics include authors,
statistical analysis used. An extraction table of all stud- location of study, objectives, design, age of partici-
ies and individual tables of relevant data from each pants, sample size, duration, variables evaluated, inter-
study was constructed. The results of the appraisals can ventions mentioned, statistical analysis used, and
be seen in the Appraisal Chart (Table II). Based on cri- outcomes. The terms gender and sex were used inter-
teria, all studies were deemed to have a high risk of changeably and were limited to "male" and "female"
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ORAL AND MAXILLOFACIAL SURGERY OOOO
4 Gabay et al. && 2024
Fig. 1. PRISMA Flow Chart for Identification of Studies. The figure illustrates the authors’ flow diagram for the selection of stud-
ies included in this systematic review using the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA).
throughout articles and therefore in this systematic were the only countries that reported swelling, 46.2%
review. and 30.8%, respectively.4 The study correlated age and
Pereira et al.4 conducted an international cross-sec- symptom treatment relationships via a chi-square test.
tional study in 2016, which included collaboration Twenty percent (20%) of people were symptomatic in
from 5 schools across 4 countries. The authors aimed the 27 to 40 year age bracket and 50% in the 40 to 50
to identify and analyze the variables of demographics, year age bracket. There were 64.3% of patients in the
clinical presentations, and radiographic findings in 50 to 60 year age bracket who were symptomatic and
patients with FOD through an international perspec- 75% of patients in the 60 to 79 year age bracket. There
tive. This study composed of 82 cases used panoramic were 92.9% of symptomatic patients who received a
and CBCT scans for proper diagnosis of FCOD in form of treatment after diagnosis. There were 57.1% of
patients. The mean age of the patients in this study was asymptomatic patients who did not receive treatment.4
53.9 years old with 99% being women and 77% being The study indicated intervention only being indicated
Black.4 Sixty-four percent (64%) of patients presented for symptomatic cases with secondary infections. Pro-
with infection at the time of diagnosis. The highest per- phylactic visits were required in combination of the
centage of patients presenting with infection was in patient maintaining good oral hygiene maintenance.
Brazil at 40.5%. There were 70.3% patients in Brazil In 2010, Alsufyani9 conducted a cross-sectional
who were symptomatic. Brazil and the United States study and literature review for the thesis defense using
Volume 00, Number 00
OOOO
Table II. Joanna Briggs Institute (JBI) critical appraisal tool
Study Inclusion criteria Subjects and setting Sample size Objective criteria Condition measured Valid methods used Were Appropriate Risk
defined? described? adequate? used for in a standard, for identification of confounding statistical of bias
measurement of reliable way for all condition? factors analysis used?
condition? participants? identified?
“Clinical Demographic Analysis Yes Yes Yes Yes, through radio- Yes Yes No Yes High
of 82 patients affected by florid graphs and clinical
osseous dysplasia: an interna- examination
tional collaborative study” Per- records
eira et al. (2016)
“Cemento-osseous dysplasia of Yes Yes Yes Yes, through radio- Yes Yes No Yes High
the jaw bones: A radiographic graphs and clinical
analysis of 118 cases” examination
Alsufyani (2010) records
“Cone-beam computed tomogra- Yes Yes Yes Yes, through radio- Yes Yes Yes Yes High
ARTICLE IN PRESS
phy analysis of cemento-osse- graphs and clinical
ous dysplasia-induced changes examination
in adjacent structures in a Bra- records
zilian population”
Kato et al. (2020)
“Periodontal and Dental Consid- Yes Yes No, using 11 cases is Yes, through radio- Yes Yes Yes Appropriate descrip- High
erations in Florid Cemento- a small sample graphs and clinical tive analysis
Osseous Dysplasia: Clinical size and therefore examination
and Radiographic Analysis of might not accu- records
11 cases” rately depict the
Martinez et al. (2019) sample frame/ tar-
get population
“Infected Cemento-Osseous Dys- Yes Yes Yes Yes, through radio- Yes Yes Yes Yes High
plasia: Analysis of 66 cases and graphs and clinical
literature review” examination
Kato et al. (2020) records
“Mandibular radiomorphometric Yes Yes Yes Yes, through radio- Yes Yes Yes Yes High
parameters of women with graphs and clinical
cemento-osseous dysplasia” examination
Kato et al. (2019) records
“Retrospective study on idio- Yes Yes No, using 20 cases is Yes, through radio- Yes Yes Yes Appropriate descrip- High
pathic bone cavity and its asso- a small sample graphs and clinical tive analysis
ciation with cementoosseous size and therefore examination
dysplasia” cannot be an accu- records
Peacock et al. (2015) rate depiction of
the sample frame/
target population
“A clinicopathologic analysis of Yes Yes Yes Yes, through radio- Yes Yes No Appropriate descrip- High
REVIEW ARTICLE
135 cases of cemento-osseous graphs and clinical tive analysis
Gabay et al. 5
dysplasia: To operate or not to examination
operate” records
Olgac et al. (2021)
Yes Yes Yes Yes Yes Yes Yes High
of bias
High
High
archives to explore clinical presentations of COD and
Risk its radiographic features. The study included records of
118 cases with patients 30 to 73 years old. The varia-
bles assessed were demographics, internal structure
analysis used?
Yes
of patients being women.9 For clinical presentation,
confounding
Yes
participants?
Yes
measurement of
examination
examination
examination
records
records
records
Yes
Yes
Yes
osseous dysplasia-associated
“Clinical and radiographic fea-
“Osseous (Cemento-osseous)
tures affected.
Chadwick et al. (2011)
“Clinical Demographic Analysis Pereira et al. (2016) DOSS The Piraciaba Dental School (1992- To correlate demographics and International 27-79 years old 82 Age; sex; gender; infection pres- Intervention only indicated for Bivariate statistical analy- 64% cases with infection reported;
of 82 patients affected by 2014); The State University of radiographic findings to FOD cross-sectional study ence; symptoms; swelling; symptomatic cases with sec- sis: Chi-square tests, 58.8% presenting with symp-
florid osseous dysplasia: an Rio de Janeiro (2005-2014); The Through an international per- stage FCOD is in ondary infections. Mainte- correlation coefficient toms, statistically insignificant;
international collaborative Head and Neck Clinical Center spective. nance of oral hygiene of Spearman, variance, 42.3% presence of swelling
study” (1997-2011); The University of required from patient coupled Tukey’s test, Cohen’s reported. Over 77.5% of patients
Pretoria in South Africa (2012- with bi-annual prophylactic kappa test with FCOD were Black women
2014); and The Texas A&M visits. and men. Mean age was
University (2003-2014) 53.9 years old. Sex found to be
statistically significant at 98.8%
(P = .0001).
“Cemento-osseous dysplasia of Alsufyani (2010) ProQuest Oral Radiology Department of the To explore clinical presentation Cross-sectional 30-73 years old 118 cases Demographics; internal structure Gold standard for diagnosing Univariate analysis; 82.9% cases were in females with
the jaw bones: A radio- University of Toronto of Cemento-osseous dyspla- study and radiopacities; periphery includes clinical and radio- Kappa Cohen’s test; median age of 42; 72.2% of
graphic analysis of 118 sia and its radiographic fea- & shape; location; effect on graphic follow-ups. Monitor- Stepwise logistic patients presented asymptom-
cases” tures. surrounding structures; diag- ing necessary for neoplastic regression; 95% confi- atic; 21.2% of cases were florid
nosis by examiner; answer or process changes. dence intervals COD; 24% had pain and swell-
examiner reported; adjusted ing; 81.4% cases in mandible
odds ratio. only; 44.9% in posterior teeth
ARTICLE IN PRESS
only. Florid COD patient at 3-
year follow up presented osteo-
myelitis. FCOD with simple
bone cysts at 2-year follow up
was more radiopaque internally,
SBC healing in one area and
enlarged in another.
“Cone-beam computed tomogra- Kato, Barra et al. PubMed Oral Medicine Clinic and the Oral To demonstrate different effects Retrospective 25-70 years old 60 COD cases Sex; age; race; presence of Follow-up indicated for lesions Descriptive analysis; Kol- 50% cases found in Black individu-
phy analysis of cemento- (2020) and Maxillofacial Radiology of cemento-osseous dysplasia cross-sectional study symptoms, swelling, infec- for treatment planning when mogorov- Smirnov als; 58 out of 60 women; 49
osseous dysplasia-induced Clinic, Universidade Federal de seen through CBCT scans. tion; stage of COD; location evaluating anatomical struc- test; Kruskal-Wallis cases diagnosed through radio-
changes in adjacent struc- Minas Gerais (2012-2018) and region of lesion; areas tures affected. test; post hoc test. graphs. 11 patients report painful
tures in a Brazilian involved; use of orthodontic Using SPSS v. 20.0. symptoms with 23 patients pres-
population” appliance present; edentulous ent with swelling. Expansion,
patients vs. dentulous. thinning, and perforation in
bucco-lingual/palatal, mesiodis-
tal, and superior-inferior areas of
cortical bone found to be statisti-
cally significant (P < .001);
8.2% of lesions infused with
tooth roots; 18% of patients pre-
sented with root resorption in
areas.
“Periodontal and Dental Consid- Martinez et al. PubMed Riber~ao Preto at the University of To demonstrate the prevalence Retrospective 40-69 years old 11 cases Age; sex; race; symptom, swell- Pulp vitality testing found to be Descriptive analysis Presence of infection reported at
erations in Florid Cemento- (2019) S~ao Paulo, Brazil (2009-2016) of FCOD with clinical fea- cross-sectional study ing and bone infection pres- indicated when there is pres- 27.3% in association with
Osseous Dysplasia: Clinical tures through radiographic ence; periodontal disease; ence of inflammation in peri- FCOD; 27.3% of patients pres-
and Radiographic Analysis of findings and to understand probing depth; bleeding on apical area. Maintenance ent with symptoms; 72.7% of 11
11 cases” presentation of symptoms. probing; clinical attachment indicated for overall oral patients were Black. Mean age
level; # teeth involved health and well-being. If of patients 53.9 years old. Sex
patient has infection, antibi- was found to be statistically sig-
otics prior to surgery may be nificant at 90.9%. Presence of
indicated. swelling and symptoms reported
at 27.3%. Average number of
teeth involved in FCOD
area = 19. Generalized periodon-
tal disease found in 72.7% and
REVIEW ARTICLE
localized in 27.3%. Biopsy
reported to be contraindicated in
Gabay et al. 7
patients with FCOD.
“Infected Cemento-Osseous Kato, Arruda et al. PubMed School of Dentistry of Universidade To describe demographic distri- Retrospective 46-68 years old 41 Florid cases Age; gender; skin color; anatom- 36 out of 41 patients with FCOD SPSS v23.0 software used Florid Cemento-Osseous Dysplasia
Dysplasia: Analysis of 66 (2020) Federal de Minas Gerais, Belo butions, manifestations, and Cross-sectional study ical location; symptom pres- received surgical curettage/ for subtype relation- found in 62.1% cases; 39 out of
cases and literature review” Horizonte, Brazil (1990-2017) treatments for infected ence; swelling presence; removal of necrotic bone. 5 ships; ANOVA test; 66 cases were female. 37 people
8 Gabay et al.
cemento-osseous dysplasia radiographic features of cases saw recurrence. Fisher’s exact test (P were not-White (P = .999); 38
patients. masses; trigger factors; treat- < .05) cases were found in the mandi-
ment; recurrence ble; 33 were symptomatic
(P = 0.155). Swelling was pres-
ent in 19 cases (P = .347); radio-
lucent and radiopaque masses
mixture features were present in
29 cases (P = .053) which was
statistically significant; 15
patients saw trigger factor being
a tooth extraction, while 7 had
trigger factor of trauma by den-
ture.
“Mandibular radiomorphometric Kato et al. (2019) PubMed School of Dentistry of Universidade To compare radiomorphometric Retrospective Mean age= 100 cases (50 with Trabecular bone in condyle, Females should be screened SPSS version 20.0 soft- Mean MCW index for group with
ORAL AND MAXILLOFACIAL SURGERY
parameters of women with Federal de Minas Gerais, Belo parameters of mandibular cross-sectional paired study 46.84 years old COD & 50 angle, molar, pre-molar, ante- more for low bone mineral ware used; McNemar- cemento-osseous dysplasia was
cemento-osseous dysplasia” Horizonte, Brazil (2014-2018) cortical bone between without COD) rior, ramus region; Mandibu- density. Patients with COD Bowker test; Wilcoxon 3.12 mm. Mean FOD for COD
women with COD and lar cortical index should be continuously moni- test; Paired t-test; patients was lower than the non-
women without COD. tored. No intervention was Cohen’s kappa test; COD group in the mandibular
stated. descriptive analysis; cortical bone region (P = .031).
Kolmogorov-Smirnov COD group had more C3 MCI
test. (P = .009)
“Retrospective study on idio- Peacock et al. PubMed Oral and Maxillofacial Pathology To assess pathological finding in Cross-sectional study 13-66 years old 20 cases Sex; age; race; location of lesion; Intervention was carried out with Descriptive analysis 11 women out of 20 patients found
pathic bone cavity and its (2015) Laboratory in Augusta Georgia patients with cemento-osse- clinical presentation; radio- surgical curettage and bleed- with COD; 90% of lesions were
association with cementooss- (2004-2014) ous dysplasia and idiopathic graphic presentation ing of stimulation. Hormonal unilocular, 50% of them being in
eous dysplasia” bone cavities. factors can affect bone women; 30% of all patients were
resorption rates based on Black women that had IBC and
demographics of patients COD together. There was 1 reoc-
involved in the study. currence; 85% of lesions were
found in the mandible.
“A clinicopathologic analysis of Olgac et al. (2021) Web of Oral and Maxillofacial Pathology To assess COD characteristics to Cross-Sectional study Mean age = 40.15 135 cases, Age; sex; localization; gross fea- Surgical intervention can be Descriptive analysis and 19 Florid COD cases with a mean
135 cases of cemento-osse- Science Department in Istanbul, Turkey aid in treatment and manage- 19 FCOD tures; type of mineralization; indicated if there is distur- data analysis using age of 40.15 years. There was a
ous dysplasia: To operate or (2005-2015) ment decisions. prominent stromal cellular bance around lesion for both Excel 2011. 5:3 ratio of females to males; 17
not to operate” pattern; radiologic features prosthetic purposes and adja- patients had symmetrical man-
cent anatomical structures. dibular pre-molar/molar lesions
Surgical intervention besides (89.5%); 16 patients had gritty
that is not indicated. Only gross features; and 11 had mod-
follow-up is. erate prominent stromal cellular
ARTICLE IN PRESS
PDL, periodontal ligament; SBC, simple bone cyst; COD, cemento-osseous dysplasia; FOD, florid osseous dysplasia; FCOD, florid cemento-osseous dysplasia; MCW, mandibular cortical width; MCI, man-
florid cemento-osseous dysplasia
Lamina dura in COD-SBCs was
< .005). Women had more soli-
or tooth movement.
visible (P < .05).
history, treatment, whether the patient was symptom-
atic, and presence of periodontal disease in FCOD
Outcomes
sis; descriptive
the time of diagnosis. However, 27.3% of patients had
significance.
analysis
signs of bone infection and 72.7% presented with gen-
eralized periodontal disease.2 The mean number of
associated solitary bone cysts
elitis develop.
Intervention
occurs.
Kato et al.8 conducted a retrospective cross-sectional
COD- associ-
subjects with
Science
Database
Web of
Web of
(2011)
(2011)
on bone mineralization.
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10 Gabay et al. && 2024
Peacock et al.12 conducted a retrospective cross-sec- showed 89% of patients had cortical bone thinning,
tional study which included records from 2004 to 16.4% of patients had lesions which involved the man-
2014. The aim of this study was to assess pathological dibular canal, 11% of patients were found to have
finding in patients with COD and idiopathic bone cavi- cysts, and 1.4% had infection at the time of diagnosis.5
ties. The variables assessed were demographics, loca- The authors did not recommend biopsies or other surgi-
tion of lesion, clinical presentations, and radiographic cal procedures in affected areas due to potential infec-
presentations. The sample size of this study was 20 tion risks. The recommendation is to maintain good
patients with a mean age of 36.3 for men and 23.2 for oral hygiene and 6-month check-ups.
women.12 Eleven females were diagnosed with COD. In 2011, Chadwick et al.14 conducted a cross-sec-
Most lesions were found in the mandible (85%). Inter- tional study which assessed and reviewed the clinical
vention was carried out with surgical curettage for presentations and radiographic findings of SBCs in
stimulation for bleeding. Hormonal factors were association with COD presence. The variables evalu-
assessed to be factors that can affect bone resorption ated in this study were lesion location, demographics,
rates based on demographics of patients involved in bone expansion presence, cortical thinning presence,
the study.12 Out of all 20 patients, only one was seen to tooth root scalloping, periodontal ligament (PDL)
have a reoccurrence. space presence, and visible lamina dura presence. Dis-
Olgac et al.13 conducted a cross-sectional study tinguishing solitary bone cysts from FCOD is crucial
which assessed COD characteristics to aid in treatment for proper diagnosis and treatment planning. The mean
and management decisions. The variables evaluated age of male subjects in this study was 18.2 years, rang-
are demographics, localization, features, type of miner- ing from 11 to 44 years old and mean of female sub-
alization, prominent stromal cellular pattern, and radio- jects was 18.8 years old, ranging from 11 to 58 years
logic features. The authors conducted a descriptive old.14 There were 68 patients observed to have SBCs
analysis after reviewing the oral biopsies and stained and 23 patients that had COD-associated SBCs. The
slides of patients. A total of 135 lesions were evaluated, authors hypothesized that in young adults, the forma-
19 of them being the FCOD subtype. Of the partici- tion of SBCs or COD-associated SBCs may be linked
pants in the study, 80% were women. The mean age of to the development and growth of the mandible and
patients with FCOD was 40.15 years. Through CBCT bone cells not being able to keep up with the growth.
scans and other radiographs, the authors determined In older adults, specifically older women, SBC devel-
radiographic features. It was determined that 16 opment may reflect the decoupling of cellular activity
patients had a mixture of radiopaque and radiolucent in the bone through a lower number of osteoblasts.14
lesions. The authors suggested that surgical interven- They emphasized the need for proper diagnosis
tion is only indicated if there are changes to anatomic between the 2 differential diagnoses in order to ensure
structures around the lesions for both prosthetic pur- proper treatment is offered to patients. However, they
poses and adjacent anatomic structures, such as with a did not state the specific type of treatment options
secondary infection. For asymptomatic COD, specifi- available for either group.
cally FCOD, only follow-up and maintenance were Alsufyani and Lam15 conducted a retrospective
recommended by the authors. The authors also recom- cross-sectional study in 2011 which aimed to assess
mended to abstain from invasive procedures, such as demographic, radiographic, and clinical features of
dental implant placement or extractions, unless abso- patients with COD. The study included 118 patients
lutely necessary. ages 30 to 58 years old. The variables analyzed were
In a 2021 retrospective cross-sectional study, Gumru demographics, clinical features, symptoms presence,
et al.5 used CBCT scans to demonstrate the importance location of lesions, surrounding structures, and mani-
of CBCT findings while exploring the demographics festations. The authors conducted a univariate and
and effects of COD on patients. The study included descriptive analysis for assessment of variables. Demo-
142 cases. The study included patients aged 24 to graphic information was known for 117 patients and
75 years. The variables assessed in this study were clinical information was available for 115 patients.
demographics, radiological characteristics, manifesta- There were 21.2% of patient archives analyzed were
tions, and subtypes of COD. There were 53.1% of FCOD lesions. There were 72.2% of cases that were
patients with the FCOD subtype who were women. asymptomatic, whereas 27.8% of patients reported
The mean age of patients with FCOD was 47 years. pain, swelling, tooth sensitivity, or tooth movement.15
There were 67.1% of patients who had FCOD in the One patient had osteomyelitis develop. The authors
mandible and 32.9% of patients had lesions in both recommended that treatment should be done conserva-
their mandible and maxilla. The region found most tively in the cases of COD unless imperative due
affected by FCOD was the posterior mandible, which to infection or differences in pathology. Invasive pro-
accounted for 97.3% of cases.5 Radiographic findings cedures, such as tooth extractions and implant
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OOOO REVIEW ARTICLE
Volume 00, Number 00 Gabay et al. 11
placements, were said to be contraindicated due to the of FCOD.5,11,14,15 As seen in Figure 3, there were 3
risk of aggravating periodontal disease or infection. studies that reported higher prevalence of FCOD
Follow-up and maintenance were recommended for in Black patients when compared with other races,
people with COD. which included White, Asian, and Hispanic.2,4,8 Con-
trastingly, Kato et al. saw an equal number of patients
DISCUSSION who were Black and other races presenting with
FCOD is a subtype of COD, presenting as multifocal FCOD.10 There were 2 studies that reported seeing
radiolucencies, mixed radiolucencies and radiopacities, FCOD more prevalent in other races than Black.12,13
and radiopacities. In later stages, masses become The varying results between studies can be attributed
increasingly radiopaque over time. FCOD is typically to the location of the study, the population demo-
diagnosed based on clinical and radiographic presenta- graphic of the region, and the access that the surround-
tions, often as incidental findings during dental visits. ing population has to these respective clinics. Peacock
FCOD often presents asymptomatically, it may become et al. also stated hormonal factors being contributors to
symptomatic spontaneously, but particularly if the area bone resorption rates based on patient demographics.
is traumatized, biopsied, or undergoes tooth extraction
due to introduction of bacteria into the bone.6 There is Radiographical presentation of FCOD
a wide range of clinical presentations of FCOD in Periapical and panoramic radiographs are usually suffi-
patients, which contributes to the management com- cient to diagnose COD and its subtypes.5 However, in
plexity for symptomatic and asymptomatic FCOD. order to understand the extent of the lesion, stage of
condition, and relationship with surrounding structures,
Geographic and demographic distributions of CBCT scans are preferred. CBCT scans show high res-
FCOD olution 3-dimensional imaging for accurate depiction
The 11 studies were conducted internationally in dif- of density of lesions, maturation stages, and character-
ferent geographic locations. Five studies were con- istics. All 11 studies included in this systematic review
ducted in Brazil.2,4,8,10,11 Two studies were conducted saw clinical examinations coupled with radiographic
in Turkey.5,13 Three studies were conducted in imaging for proper diagnosis of FCOD. Three studies
Canada,9,14,15 whereas only one study was conducted used only panoramic radiographs for radiographic
in the United States.12 All literature included in this diagnosis.2,4,12 Kato et al. used panoramic radiographs
systematic review were cross-sectional design studies coupled with periapical radiographs for diagnosis,
that used secondary data from archives of radiographic whereas Gumru et al. used panoramic radiographs
imaging from their respective universities. Due to the and CBCT scans. In contrast, the remaining 6 studies
nature of the cross-sectional design, all had high risk of all used combinations of periapical, panoramic,
bias. and CBCT imaging for accurate and definite
The age range of patients for the majority of studies diagnosis.9,10,11,13 15 Although every study used pan-
included in this systematic review was similar, being oramic radiographs with clinical examinations for
25 to 70 years old. The mean age between studies was FCOD diagnosis, literature called on using CBCT
40 years old. Two studies had outliers in age demo- scans for the most precise evaluation.
graphics, both having 2 youths, aged 13 and 11, respec-
tively, included in their studies.12,14 Although the age Prevalence of FCOD by region of the oral cavity
ranges in these studies can pose as confounding factors, affected
the average age of patients in both studies were never- All 11 studies saw a higher frequency of FCOD occur-
theless similar to the rest of the studies, therefore ring in the mandibular region of the jaw. Kato et al.
inconsequential to the systematic review. Pereira et al. found 60 cases of FCOD in the mandible and 24 cases
reported significance of increasing age correlating to in the maxillary and mandibular regions.11 Similarly,
an increased number of symptomatic patients.4 Martinez et al. observed FCOD in the maxillary and
Throughout the studies, demographics proved to be mandibular regions.2 Gumru et al. found FCOD in the
valuable variables by allowing us to understand how mandible in 67.1% of patients and in the maxillary and
the disease trends. The variables of age, race, and gen- mandible in 16.99% of patients.5 Kato et al. (2019)
der are also confounding factors that influence FCOD found that the highest frequency of FCOD was present
and have an effect on the disease. Throughout all 11 in the mandibular anterior region. Similarly, Peacock
studies, there was a significant association between et al. found the condition in the mandibular anterior
gender and FCOD. As seen in Figure 2, all 11 studies and posterior region.12 Alsufyani found most cases in
showed female patients had higher prevalence with the mandibular posterior region and 16 cases unilater-
FCOD than male patients. Five studies did not report ally in the maxillary and mandibular regions.9 Alsu-
any prevalence or correlation of race to the diagnosis fyani and Lam found presentations largely bilaterally
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Fig. 2. Association of gender demographics and FCOD between studies. The figure depicts the findings for gender demographics
over the 11 studies. FCOD, florid cemento-osseous dysplasia.
in the mandible, but also in all 4 quadrants.15 Three al. reported 23 patients with swelling and 11 patients
studies primarily saw patients presenting with FCOD with symptoms.9,10 However, these 2 studies did not
in the mandibular region.8,13,14 report patients with infection. Kato et al. reported 19
patients with swelling and 33 patients with symptoms,
Clinical presentations of symptomatic versus whereas Alsufyani and Lam reported swelling in 32
asymptomatic FCOD patients and symptoms in 35 patients.8,15 However,
As defined in this systematic review, symptomatic these 2 studies also did not report patients with infec-
FCOD included presence of swelling, symptoms, infec- tion. Peacock et al. only reported symptoms in 5
tion, or pain. As seen in Figure 4, clinical presentations patients, whereas Gumru et al. reported symptoms in 8
of FCOD were differentiated into 3 categories of pres- patients and 1 patient presenting with infection.5,12,
ence of swelling, symptoms, and infection. Pereira et Neither study reported the presence of swelling. Con-
al. reported 22 patients with swelling, 30 patients with trastingly, 3 studies did not report swelling, symptoms,
symptoms, and 18 patients with infected FCOD, or infection.11,13,14 Through these findings and as seen
whereas Martinez et al. reported 3 patients with swell- in Figure 4, the most common clinical presentation was
ing, symptoms, and infection during clinical and radio- painful, symptomatic FCOD, followed by swelling in
graphic examinations.2,4 Alsufyani reported 28 patients affected areas of the jaw, and, last, the presence of
with swelling and 33 with symptoms, whereas Kato et infected structures in FCOD regions.
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ARTICLE IN PRESS
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Fig. 4. Clinical presentations of FCOD across studies. The figure 4 exemplifies the clinical presentations of FCOD as being dif-
Fig. 3. Racial distribution of FCOD across studies. The figure depicts the findings for racial distributions across the 11 studies.
Gabay et al. 13
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Fig. 5. Treatment options for symptomatic FCOD recommended across studies. The figure presents the treatment options for
symptomatic FCOD. FCOD, florid cemento-osseous dysplasia.
Treatment recommendations of symptomatic vs antibiotic treatment as the primary course for symp-
asymptomatic FCOD tomatic FCOD.8 However, all studies that stated that if
Across studies, the foremost treatment recommenda- surgical intervention was carried out, antibiotic therapy
tion for symptomatic FCOD was monitoring and pro- should be used post-treatment.
phylaxis at biannual check-ups with a combination of Nine studies recommended monitoring the progres-
maintenance of good oral hygiene. This is depicted in sion of condition. However, the remaining 2 studies
Figure 5. For symptomatic FCOD, 5 studies recom- did not highlight this treatment option in their
mended a surgical treatment option.2,4,8,12,13 Whereas work.11,14 Nine studies suggested biannual prophylaxis
Pereira et al. recommended intervention only for sec- as upkeep and means of maintaining proper oral
ondary infections in symptomatic cases, Olgac et al. hygiene to avoid infection. However, 2 studies did not
only recommended surgical intervention when there recommend prophylaxis treatment11,14 and listed no
was presence of anatomic changes around the lesion specific interventions.
and other surrounding anatomic structures.4,13 Marti-
nez et al. recommended pulp vitality testing to be con- STRENGTHS AND LIMITATIONS OF STUDY
ducted in inflamed periapical areas of FCOD affected This systematic review was the first review conducted
regions.2 Kato et al. recommended surgical removal of on the optimal management strategies of FCOD. How-
necrotic bone in affected area by surgical curettage.8 In ever, it was challenging for the authors to conduct this
contrast, Peacock et al. recommended surgical inter- systematic review without a sufficient quantity of well-
vention through surgical curettage for stimulation of designed and controlled studies on treatment options of
bleeding for symptomatic and asymptomatic FCOD.12 FCOD which were not case reports. Multiple studies
As seen in Figure 6, all other studies did not recom- included in this review also had smaller patient pools
mend surgical intervention for asymptomatic FCOD. which might not be reflective of the population.
Kato et al. was the only study that recommended Another limitation of this systematic review and gap in
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Volume 00, Number 00 Gabay et al. 15
Fig. 6. Treatment options for asymptomatic FCOD recommended across studies. The figure presents the treatment options for
asymptomatic FCOD. FCOD, florid cemento-osseous dysplasia.
literature was that studies including pulp vitality testing symptomatic cases, and another study recommended
did not include negative pulp vitality tests or directions surgical intervention only when there was presence of
for what to do in the case of a negative pulp vitality changes to anatomic structures around the lesion and
test. There was also an inability to complete a meta- other surroundings. One study recommended pulp
analysis due to the nature of the qualitative data pri- vitality testing to be conducted in the periapical areas
marily presented in the studies. of affected regions.2 Another study recommended sur-
gical removal of necrotic bone in the affected area by
Future direction surgical curettage.8 Contrastingly, another study rec-
Randomized control trials (RCTs) for treatment of ommended surgical intervention through surgical
FCOD can further provide insight on definitive optimal curettage for stimulation of bleeding for symptomatic
recommended therapy and management. Further evalu- and asymptomatic FCOD.12 For cases of asymptomatic
ations should be conducted to determine how genetics FCOD, prophylaxis and monitoring were the most
and/ or hormones may influence FCOD. Additional common treatment options followed by no interven-
evaluations of the medical history can also be con- tion. Only one study recommended surgical treatment
ducted to understand possible linkage and correlations for asymptomatic FCOD. Nine studies suggested bian-
of FCOD to pre-existing conditions. nual prophylaxis for both symptomatic and asymptom-
atic FCOD as upkeep and a means of maintaining
CONCLUSION proper oral hygiene to avoid infection. In cases of sec-
In conclusion, 11 cross-sectional international studies ondary infection or surgical intervention carried out,
conducted across Canada, the United States, Brazil, antibiotic prophylaxis must be used.
and Turkey were analyzed and included in this system- A majority of publications support the main manage-
atic review with meta review. Eight studies reported ment strategy for asymptomatic FCOD being good oral
symptoms as the clinical presentation of FCOD, mak- hygiene maintenance through biannual dental prophy-
ing it the most common. Six studies reported swelling laxis and radiographs for change evaluation. Existing
upon clinical presentation. The least common clinical literature tends to support the usage of surgical inter-
presentation was infection, with only one study report- vention through curettage with stimulation of bleeding,
ing. For symptomatic FCOD, 5 studies recommended a or pulp vitality testing of specific areas with periapical
surgical treatment option. One study recommended inflammation for symptomatic FCOD, but to avoid sur-
intervention only for secondary infections in gical intervention for asymptomatic FCOD. After
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16 Gabay et al. && 2024
surgical intervention, continued dental prophylaxis and cemento-osseous dysplasia: clinical and radiographic analysis of
monitoring are the most common treatment options 11 cases. Oral Health Prev Dent. 2019;17(5):425-431. https://
suggested by clinicians. doi.org/10.3290/j.ohpd.a43273.
3. Ogunsalu CO, Lewis A, Doonquah L. Benign fibro-osseous
lesions of the jaw bones in Jamaica: analysis of 32 cases. Oral
CONFLICT OF INTEREST STATEMENT Dis. 2001;7(3):155-162.
None. 4. Pereira DL, Pires FR, Lopes MA, et al. Clinical, demographic,
and radiographic analysis of 82 patients affected by florid osse-
CREDIT AUTHORSHIP CONTRIBUTION ous dysplasia: an international collaborative study. Oral Surg
STATEMENT Oral Med Oral Pathol and Oral Radiol. 2016;122(2):250-257.
5. Gumru B, Akkitap MP, Deveci S, Idman E. A retrospective cone
Miriam Gabay: Writing review & editing, Writ-
beam computed tomography analysis of cemento-osseous dys-
ing original draft, Resources, Methodology, Formal plasia. J Dental Sci. 2021;16(4):1154-1161.
analysis, Data curation, Conceptualization. Louis 6. MacDonald-Jankowski DS. Florid cemento-osseous dysplasia: a
DiPede: Writing review & editing, Writing origi- systematic review. Dentomaxillofac Radiol. 2003;32(3):141-149.
nal draft, Supervision, Methodology, Conceptualiza- 7. MacDonald D, ed. Oral and maxillofacial radiology: a diagnos-
tic approach, John Wiley & Sons; 2019.
tion. Maria Fornatora: Writing review & editing,
8. Kato CD, de Arruda JA, Mendes PA, et al. Infected cemento-
Methodology, Conceptualization. Jie Yang: Writing osseous dysplasia: analysis of 66 cases and literature review.
review & editing, Methodology, Conceptualization. Head Neck Pathol. 2020;14:173-182.
Chukwuebuka Ogwo: Writing review & editing, 9. Alsufyani NA. Cemento-osseous Dysplasia of the Jaw Bones: A
Methodology, Formal analysis, Conceptualization. Radiographic Analysis of 118 Cases (Doctoral dissertation).
2010.
10. Kato CD, Barra SG, Amaral TM, et al. Cone-beam computed
ACKNOWLEDGMENTS tomography analysis of cemento-osseous dysplasia-induced
Presentation changes in adjacent structures in a Brazilian population. Clin
This systematic review was previously presented by Oral Investig. 2020;24:2899-2908.
Miriam Gabay at the American Association for Dental, 11. Kato CN, Barra SG, Pereira MJ, et al. Mandibular radiomor-
phometric parameters of women with cemento-osseous dys-
Oral, and Craniofacial Research (AADOCR) confer-
plasia. Dentomaxillofac Radiol. 2019;49(4):20190359.
ence in Portland, Oregon, on March 15, 2023. 12. Peacock ME, Krishna R, Gustin JW, Stevens MR, Arce RM,
Abdelsayed RA. Retrospective study on idiopathic bone cavity
FUNDING SOURCES and its association with cementoosseous dysplasia. Oral Surg
This research did not receive any specific grant from Oral Med Oral Pathol Oral Radiol. 2015;119(4). e246-51.
13. Olgac V, Sinanoglu A, Selvi F, Soluk-Tekkesin ME. A clinico-
funding agencies in the public, commercial, or not-for- pathologic analysis of 135 cases of cemento-osseous dysplasia:
profit sectors. There were no commercial associations, To operate or not to operate? J Stomatol Oral Maxillofac Surg.
current and within the past 5 years, that might pose a 2021;122(3):278-282.
potential perceived, or real conflict of interest. 14. Chadwick JW, Alsufyani NA, Lam EW. Clinical and radio-
graphic features of solitary and cemento-osseous dysplasia-asso-
REFERENCES ciated simple bone cysts. Dentomaxillofac Radiol. 2011;40
1. Slootweg P, Slootweg P. Fibro-osseous lesions. Pathology of the (4):230-235.
Maxillofacial Bones: A Guide to Diagnosis. 2015:123-156. 15. Alsufyani NA, Lam EW. Cemento-osseous dysplasia of the jaw
2. de Jesus Hernandez Martinez C, Vargas Villafuerte KR, Felix bones: key radiographic features. Dentomaxillofac Radiol.
Silva PH, et al. Periodontal and dental considerations in florid 2011;40(3):141-146.