BACKGROUND: Since there are many differential diagnoses for cemento-osseous dysplasia (COD), it is very difficult for dentists to avoid misdiagnosis. In particular, if COD is related to an embedded tooth, differential diagnosis is difficult. However, there have been no reports on the characteristics of the imaging findings of COD associated with embedded teeth. The aim of the present study was to investigate the occurrence and imaging characteristics of cemento-osseous dysplasia (COD) associated with embedded teeth, in order to appropriately diagnose COD with embedded teeth.
METHODS: The radiographs with or without histological findings of 225 patients with COD were retrospectively analyzed. A retrospective search through the picture archiving and communication system (PACS) of the Division of Oral and Maxillofacial Radiology of Kyushu Dental University Hospital was performed to identify patients with COD between 2011 and 2022.
RESULTS: Fifteen COD-associated embedded mandibular third molars were identified in 13 patients. All 13 patients were asymptomatic. On imaging, COD associated with embedded mandibular third molars appeared as masses that included calcifications around the apex of the tooth. On panoramic tomography, COD showed inconspicuous internal calcification similar to that of odontogenic cysts or simple bone cysts, especially in patients with COD only around the mandibular third molar region. Those with prominent calcification resembled cemento-ossifying fibroma, calcifying epithelial odontogenic tumor, calcifying odontogenic cyst, adenomatoid odontogenic tumor, and so on, as categories of masses that include calcifications on panoramic tomography and computed tomography.
CONCLUSIONS: The current investigation is the first to report and analyze the imaging characteristics of COD associated with embedded teeth. It is important to consider the differences between COD and other cystic lesions on panoramic tomography, and the differences between COD and masses that include calcifications on CT.

Cemento-ossifying fibroma (COF) and juvenile ossifying fibroma (JOF) have been considered distinct entities within the category of fibro-osseous lesions. This study aimed to assess osteoblast and osteoclast activity in COF and JOF by investigating bone resorption markers, specifically receptor activator of nuclear factor-kB (RANK), RANK ligand (RANKL), and its inhibitor osteoprotegerin (OPG). A comparative analysis of these markers was performed on all lesions. Immunohistochemistry was employed to evaluate and quantify the expression of these biomarkers in a sample of 20 cases of cemento-ossifying fibroma (COF), 15 cases of psammomatoid juvenile ossifying fibroma (PsJOF), and 10 cases of trabecular juvenile ossifying fibroma (TrJOF). The expression of osteoprotegerin was significantly higher in cemento-ossifying fibroma (33.9±13.0) compared to trabecular juvenile ossifying fibroma (27.3±9.2) and psammatoid ossifying fibroma (25.2±14.9), with the COF showing the highest expression followed by the latter two (p=0.037). There was a higher percentage (80%) of stromal fibroblast cells that showed positive expression of RANKL in cemento-ossifying fibroma (COF) compared to psammomatoid juvenile ossifying fibroma (PsJOF) (33.3%) and trabecular juvenile ossifying fibroma (TrJOF) (30.0%) when considering a positive expression score of 3 (p=0.024). Cemento-ossifying fibroma demonstrated the highest expression of osteoprotegerin and RANKL-positive stromal fibroblast cells, followed by psammomatoid juvenile ossifying fibroma and trabecular juvenile ossifying fibroma. These findings provide valuable insights into the pathogenesis of these lesions.

BACKGROUND: Odontogenic tumours are infrequent lesions. Studies on the frequency of odontogenic tumours from Latin America are scarce. This work aimed to determine the relative frequency of odontogenic tumours in a Chilean population using the 2022 World Health Organization classification.
METHODS: This is a case series retrospective study. We reviewed 35,530 samples from 1975 to 2022 from the Oral Pathology Referral Institute and the Pathological Anatomy Service, Faculty of Dentistry, University of Chile. We utilized the 2022 World Health Organization classification for histological typification.
RESULTS: According to 2022 World Health Organization classification, 544 odontogenic tumours were confirmed. The most frequent odontogenic tumours were: odontoma (n=241; 44.3%), ameloblastoma (n=109; 20.0%) and cemento-ossifying fibroma (n=71; 13.1%). Benign odontogenic tumours corresponded to 538 cases (98.9%) and malignant tumours were only six cases (1.1%).
CONCLUSIONS: In our population, odontoma was the most frequent odontogenic tumour followed by ameloblastoma and cemento-ossifying fibroma. Malignant odontogenic tumours were very rare. The results of this study are similar to reports from America, but there are some differences concerning the data from Africa and Asia.

BACKGROUND: Familial gigantiform cementoma (FGC) is a rare tumor characterized by the early onset of multi-quadrant fibro-osseous lesions in the jaws, causing severe maxillofacial deformities. Its clinicopathological features overlap with those of other benign fibro-osseous lesions. FGC eventually exhibits progressively rapid growth, but no suspected causative gene has been identified.
METHODS: In this study, three patients with FGC were recruited, and genomic DNA from the tumor tissue and peripheral blood was extracted for whole-exome sequencing.
RESULTS: Results showed that all three patients harbored the heterozygous mutation c.1067G > A (p.Cys356Tyr) in the ANO5 gene. Furthermore, autosomal dominant mutations in ANO5 at this locus have been identified in patients with gnathodiaphyseal dysplasia (GDD) and are considered a potential causative agent, suggesting a genetic association between FGC and GDD. In addition, multifocal fibrous bone lesions with similar clinical presentations were detected, including five cases of florid cemento-osseous dysplasia, five cases of polyostotic fibrous dysplasia, and eight cases of juvenile ossifying fibromas; however, none of them harbored mutations in the ANO5 gene.
CONCLUSIONS: Our findings indicate that FGC may be an atypical variant of GDD, providing evidence for the feasibility of ANO5 gene testing as an auxiliary diagnostic method for complex cases with multiple quadrants.

BACKGROUND: Fibrous dysplasia (FD) and cemento-ossifying fibroma (COF) are the most common gnathic fibro-osseous lesions. These diseases exhibit remarkable overlap of several clinicopathological aspects, and differential diagnosis depends on the combination of histopathological, radiographic, and clinical aspects. Their molecular landscape remains poorly characterized, and herein, we assessed their proteomic and phosphoproteomic profiles.
METHODS: The quantitative differences in protein profile of FD and COF were assessed by proteomic and phosphoproteomic analyses of formalin-fixed paraffin-embedded tissue samples. Pathway enrichment analyses with differentially regulated proteins were performed.
RESULTS: FD and COF exhibited differential regulation of pathways related to extracellular matrix organization, cell adhesion, and platelet and erythrocytes activities. Additionally, these lesions demonstrated distinct abundance of proteins involved in osteoblastic differentiation and tumorigenesis and differential abundance of phosphorylation of Ser61 of Yes-associated protein 1 (YAP1).
CONCLUSIONS: In summary, despite the morphological similarity between these diseases, our results demonstrated that COF and DF present numerous quantitative differences in their proteomic profiles. These findings suggest that these fibro-osseous lesions trigger distinct molecular mechanisms during their pathogenesis. Moreover, some proteins identified in our analysis could serve as potential biomarkers for differential diagnosis of these diseases after further validation.

UNASSIGNED: The clinical manifestations of these lesions comprises pain, paresthesia, swelling, drainage, tooth loss, root resorption, and facial deformity. Alteration in oral and maxillofacial (OMF) tissues of the lesions may cause esthetically and functionally unfavourable effects in patients.
UNASSIGNED: To determine the frequency of odontogenic cysts, tumors, and other lesions.
UNASSIGNED: Patient\'s records of histopathological reports from the archives of the Department of Oral and Maxillofacial Pathology were obtained and reviewed over a period of 2 years, and therefore, the lesions were classified into four groups. In cases of recurrent lesions, only the primary diagnosis was considered. The research protocol was approved by the ethical committee of the institution.
UNASSIGNED: About 56.4% of males had ameloblastoma, 54.9% of females had cementoma. 59.4% females had giant cell granuloma, 87.5% females had pyogenic granuloma, 77.2% females had osteoma, 65.1% of the female population were belongs to the benign fibro-osseous lesions and 50.2% of females were from fibrous dysplasia in group 3. About 58.8% females had squamous cell carcinoma in group 4.
UNASSIGNED: To adequately determine the prevalence and incidence rate of OMF lesions, biopsies performed by other specialists such as otolaryngologists and plastic surgeons in OMF regions should also be evaluated.

BACKGROUND: Benign neoplasms exhibit most of the cellular phenomena considered hallmarks of cancer, except the capacity to metastasize. Thus, the elucidation of the mechanisms associated with the progression of benign neoplasms may complement and clarify the mechanisms involved in carcinogenesis. Benign odontogenic tumours often result in facial deformities and morbidities, and have complex pathogenesis, mainly due to the diversity of interactions between the odontogenic epithelium and the ectomesenchyme. Primary cell culture of such tumours is not only difficult to be established and maintained, but also tumour cells lose characteristic cellular morphology. Considering gene expression, growth, migration, proliferation and cellular morphology are controlled by cell-cell interactions and cell-extracellular matrix interactions, cell culture in 3D substrates has gained space as a way to overcome some of the limitations of traditional monolayer cell culture systems.
METHODS: In this study, fragments obtained from mesenchymal odontogenic tumours were cultured in type I collagen scaffolds. Invasion tests were performed in these models, as well as phenotypic characterization of the cultured tumours.
RESULTS: The results obtained for the odontogenic myxoma and the cemento-ossifying fibroma demonstrate a good reproduction of the growth pattern of these tumours under ex vivo conditions. Microscopic evaluation showed maintenance of cell viability in the explants for more than 30 days, without the presence of necrosis.
CONCLUSIONS: This is the first study involving long-term 3D primary cultures of benign odontogenic tumours, which is expected to support complex approaches to cell and molecular biology, and to serve as an experimental model for testing molecular therapies.

The aims of this study were to describe the clinical and radiologic features of 383 fibro-osseous lesions (FOLs) from an oral pathology reference service in Brazil and to compare the findings with previous studies. The hypothesis of the study was that the most common type of FOL would differ from other investigations.
We conducted a descriptive and retrospective study with review of the records of the clinical and biopsy services (1990 to 2015). All records of the patients included showed a definitive diagnosis of FOL. The primary outcome variable was the type of FOL, and the predictor variables were gender, age, ethnicity, location, and clinical and radiologic characteristics. Descriptive analyses and χ2 tests were performed. The P value was set at .05.
From the 27,998 records available, 383 showed FOLs, with 187 (48.8%) being cemento-osseous dysplasias (CODs), 103 (26.9%) being fibrous dysplasias (FDs), and 93 (24.3%) being ossifying fibromas (OFs). The mean age of the patients was 38.5 ± 17.5 years. CODs presented a predilection for female gender (n = 314, 82.0%), African descent (n = 134, 71.6%), and the mandible (n = 248, 64.6%). The most common radiologic feature was a mixed radiolucent-radiopaque image (n = 149, 51.7%). FDs and OFs were commonly diagnosed during the first and second decades of life (P < .001), whereas CODs were more frequently diagnosed beyond the third decade (P < .001). Secondary osteomyelitis was more significantly observed in CODs (P < .001) than in FDs and OFs. Swelling was more frequently reported for FDs and OFs than for CODs (P < .001).
CODs were the most frequently observed FOLs in this Brazilian population. Female patients, patients of African descent, and patients with mandibular localization were most commonly affected by these conditions. The most common type of FOL differed from that in similar case reports or series from various geographic locations. It is believed that the data source (clinical and/or biopsy services) can directly influence the outcome.

BACKGROUND: Fibro-osseous lesions, a diverse group of bone disorders including developmental, reactive or dysplastic diseases and neoplasms, share overlapping clinical, radiographic and histopathologic features and demonstrate a wide range of biological behaviour.
OBJECTIVE: To evaluate the characteristics, treatment and outcome of benign fibro-osseous lesions of the jaws in children.
METHODS: All patients with fibro-osseous lesions of the jaws treated at the department of Oral and Maxillofacial Surgery of the «A & P Kyriakou» Children\'s Hospital of Athens from 2000 to 2011 were included in this study. Data were retrieved from patients\' files and their present situation was registered.
RESULTS: Sixteen males and 10 females (mean age 8.5 years) were treated. Fibrous dysplasia was most often encountered (26.9%), and the mandible was the most frequent location (76.9%). All cases were surgically treated and histopathologically confirmed. Marginal ostectomy was performed in 7 cases, partial ostectomy in 4, enucleation and curettage in 10 and trimming-remodelling in 5 cases. Mean follow-up was of 5.5 years with no recurrence, except in one case of fibrous dysplasia.
CONCLUSIONS: Fibro-osseous lesions, although sharing similar microscopic features, exhibit a variety of clinical behaviour rendering their treatment highly individualized.

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