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Asian Pacific Journal of Cancer... Aug 2023Primordial odontogenic tumor is a rare odontogenic tumor reported for the first time in 2014. It was included in the latest edition of World Health Organization... (Review)
Review
BACKGROUND AND OBJECTIVE
Primordial odontogenic tumor is a rare odontogenic tumor reported for the first time in 2014. It was included in the latest edition of World Health Organization classification of Head and Neck Tumors as a new benign mixed epithelial-mesenchymal neoplasm. To date, 26 cases has been reported in literature. The aim of this study was to determine the possible presence of primordial odontogenic tumor in the previously diagnosed cases with myxoid stroma.
MATERIALS AND METHODS
This study was a retrospective descriptive study that was concluded in Oral and Maxillofacial Pathology Department, School of Dentistry, Tehran University of Medical Sciences. We reviewed all 19,380 cases from 19,66 to find the lesions with myxoid stroma which were in differential diagnosis with primordial odontogenic tumor. These cases should be associated with an impacted or unerupted tooth and belong to the patients under 20 years old.
RESULTS
We find 503 pericoronal lesions with myxoid stroma. Three cases were isolated after recut and reevaluation for finding fulfill histologic features. After immunohistochemical analysis, we find a new case of developing primordial odontogenic tumor associated with odontoma.
DISCUSSION
Primordial odontogenic tumor a recently described odontogenic tumor has a well-defined clinicopathological and immunohistochemical profile and should be differentiated from the others pericoronal lesions. It is the first time that this archival review has been done to find probable cases of primordial odontogenic tumor.
Topics: Humans; Young Adult; Adult; Retrospective Studies; Iran; Odontogenic Tumors; Diagnosis, Differential; Neoplasms, Glandular and Epithelial
PubMed: 37642073
DOI: 10.31557/APJCP.2023.24.8.2845 -
La Revue Du Praticien May 2024
Topics: Humans; Ameloblastoma; Jaw Neoplasms
PubMed: 38833232
DOI: No ID Found -
Oral Surgery, Oral Medicine, Oral... Mar 2024To evaluate the diagnostic capability of artificial intelligence (AI) for detecting and classifying odontogenic cysts and tumors, with special emphasis on odontogenic... (Review)
Review
OBJECTIVE
To evaluate the diagnostic capability of artificial intelligence (AI) for detecting and classifying odontogenic cysts and tumors, with special emphasis on odontogenic keratocyst (OKC) and ameloblastoma.
STUDY DESIGN
Nine electronic databases and the gray literature were examined. Human-based studies using AI algorithms to detect or classify odontogenic cysts and tumors by using panoramic radiographs or CBCT were included. Diagnostic tests were evaluated, and a meta-analysis was performed for classifying OKCs and ameloblastomas. Heterogeneity, risk of bias, and certainty of evidence were evaluated.
RESULTS
Twelve studies concluded that AI is a promising tool for the detection and/or classification of lesions, producing high diagnostic test values. Three articles assessed the sensitivity of convolutional neural networks in classifying similar lesions using panoramic radiographs, specifically OKC and ameloblastoma. The accuracy was 0.893 (95% CI 0.832-0.954). AI applied to cone beam computed tomography produced superior accuracy based on only 4 studies. The results revealed heterogeneity in the models used, variations in imaging examinations, and discrepancies in the presentation of metrics.
CONCLUSION
AI tools exhibited a relatively high level of accuracy in detecting and classifying OKC and ameloblastoma. Panoramic radiography appears to be an accurate method for AI-based classification of these lesions, albeit with a low level of certainty. The accuracy of CBCT model data appears to be high and promising, although with limited available data.
PubMed: 38845306
DOI: 10.1016/j.oooo.2024.03.004 -
Head and Neck Pathology Sep 2023Odontogenic carcinosarcoma (OCS) is a rare odontogenic malignancy with limited characterization and unexplored molecular features. We report clinicopathologic and... (Review)
Review
BACKGROUND
Odontogenic carcinosarcoma (OCS) is a rare odontogenic malignancy with limited characterization and unexplored molecular features. We report clinicopathologic and molecular findings in 3 additional OCS and review the literature.
METHODS
3 OCS (5.1%) were identified among 59 malignant odontogenic tumors (in our archives from 1992 to 2022). Clinical, radiologic, histopathologic, immunophenotypic, and molecular findings were reviewed. Data from prior case reports and systematic or non-systematic reviews were extracted for analysis.
RESULTS
Three mandibular OCS (age range: 66 to 72 years; 1 male, 2 females) were identified. Case 1 had novel clear-cell morphology, multiple recurrences, and a lethal outcome 28 months after resection. EWSR1 rearrangements were negative, but the tumor showed focal nuclear β-catenin and strong LEF-1 immunoreactivity. Case 2 demonstrated ameloblastic and sclerosing features and encased the inferior alveolar nerve; the patient was disease-free 22 months after resection with adjuvant chemoradiation therapy. LEF-1 was again strongly positive, and next-generation sequencing demonstrated 9p region-(CDKN2A, CDKN2B) copy number loss, and 12q region-(MDM2, CDK4) copy number gain. Case 3 showed clear-cell and markedly sclerosing features; no follow-up information was available. Literature review along with the current cases yielded 20 cases. OCS showed a male predilection (1.5:1), mandibular predominance (80%, typically posterior), and a bimodal age distribution (modes: 27.7 years, 62.7 years). OCS presented as masses (100%), often with pain (55%), and paresthesia (45%). Tumors were typically radiolucent (88.9%), with bone destruction (61.1%), and/or tooth effacement (27.8%). Preoperative biopsy was sensitive for malignancy (85.7%). At least 45% show evidence for a precursor lesion. 3-year DSS and DFS were 58% and 35%, respectively. Regional and distant (usually lung) metastatic rates were 25% and 31.3%, respectively. Increased mitotic rates and presence of tumor necrosis trended toward worse DSS and DFS.
CONCLUSION
OCS is a rare but aggressive malignancy, often arising from precursor tumors and may represent a terminal phenotype rather than a distinct entity. We describe novel clear-cell and sclerosing morphologies. Wnt pathway alterations appear important. Mitotic rates and necrosis may be adverse prognosticators. In keeping with nomenclature trends in other sites, OCS may be more appropriately designated as "biphasic sarcomatoid odontogenic carcinomas."
Topics: Female; Humans; Male; Aged; Adult; Odontogenic Tumors; Mouth Neoplasms; Carcinosarcoma; Carcinoma; Biopsy
PubMed: 37486533
DOI: 10.1007/s12105-023-01569-3 -
Modern Pathology : An Official Journal... Jul 2023Sinonasal myxoma (SNM) is a rare benign mesenchymal tumor that arises in the sinonasal cavity or maxilla and almost exclusively affects young children. Currently, it is...
Sinonasal myxoma (SNM) is a rare benign mesenchymal tumor that arises in the sinonasal cavity or maxilla and almost exclusively affects young children. Currently, it is considered a specific entity, but its molecular characteristics have not been reported. Lesions diagnosed as SNM and odontogenic myxoma/fibromyxoma were identified from the participating institutions, and the clinicopathologic features were recorded. Immunohistochemistry for β-catenin was performed in all cases with available tissue. Next-generation sequencing was performed in all cases with SNM. Five patients with SNM were identified, including 3 boys and 2 girls with an age range of 20-36 months (mean: 26 months). The tumors were well defined, centered in the maxillary sinus, surrounded by a rim of woven bone, and composed of a moderately cellular proliferation of spindle cells oriented in intersecting fascicles in a variably myxocollagenous stroma that contained extravasated erythrocytes. Histologically, the tumors resembled myxoid desmoid fibromatosis. Three tested cases showed nuclear expression of β-catenin. In 3 tumors, next-generation sequencing revealed intragenic deletions of APC exons 5-6, 9 and 15, or 16, respectively, with concurrent loss of the other wild-type copy of APC predicted to result in biallelic inactivation. The deletions were identical to those that occur in desmoid fibromatosis, and copy number analysis raised the possibility that they were germline. In addition, 1 case showed the possible deletion of APC exons 12-14, and another case exhibited a CTNNB1 p. S33C mutation. Ten patients with odontogenic myxoma/fibromyxoma were identified, including 4 women and 6 men (mean age: 42 years). Seven tumors involved the mandible and 3 the maxilla. Histologically, the tumors differed from SNM, and all cases lacked nuclear expression of β-catenin. These findings suggest that SNM represents a myxoid variant of desmoid fibromatosis that often arises in the maxilla. The APC alterations might be germline, and therefore, genetic testing of the affected patients should be considered.
Topics: Child; Male; Humans; Female; Child, Preschool; Infant; Adult; Fibromatosis, Aggressive; beta Catenin; Mutation; Genetic Testing; Exons
PubMed: 37059229
DOI: 10.1016/j.modpat.2023.100189 -
Genes Aug 2023Stem cells have been associated with self-renewing and plasticity and have been investigated in various odontogenic lesions in association with their pathogenesis and... (Review)
Review
BACKGROUND
Stem cells have been associated with self-renewing and plasticity and have been investigated in various odontogenic lesions in association with their pathogenesis and biological behavior. We aim to provide a systematic review of stem cell markers' expression in odontogenic tumors and cysts.
METHODS
The literature was searched through the MEDLINE/PubMed, EMBASE via OVID, Web of Science, and CINHAL via EBSCO databases for original studies evaluating stem cell markers' expression in different odontogenic tumors/cysts, or an odontogenic disease group and a control group. The studies' risk of bias (RoB) was assessed via a Joanna Briggs Institute Critical Appraisal Tool. Meta-analysis was conducted for markers evaluated in the same pair of odontogenic tumors/cysts in at least two studies.
RESULTS
29 studies reported the expression of stem cell markers, e.g., SOX2, OCT4, NANOG, CD44, ALDH1, BMI1, and CD105, in various odontogenic lesions, through immunohistochemistry/immunofluorescence, polymerase chain reaction, flow cytometry, microarrays, and RNA-sequencing. Low, moderate, and high RoBs were observed in seven, nine, and thirteen studies, respectively. Meta-analysis revealed a remarkable discriminative ability of SOX2 for ameloblastic carcinomas or odontogenic keratocysts over ameloblastomas.
CONCLUSION
Stem cells might be linked to the pathogenesis and clinical behavior of odontogenic pathologies and represent a potential target for future individualized therapies.
PubMed: 37761874
DOI: 10.3390/genes14091735 -
Medicina Oral, Patologia Oral Y Cirugia... Nov 2023Odontogenic tumours are infrequent lesions. Studies on the frequency of odontogenic tumours from Latin America are scarce. This work aimed to determine the relative... (Review)
Review
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.
MATERIAL AND 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.
Topics: Humans; Ameloblastoma; Odontoma; Retrospective Studies; Cementoma; Chile; Odontogenic Tumors; World Health Organization
PubMed: 37823289
DOI: 10.4317/medoral.26008 -
Journal of Oral Pathology & Medicine :... Sep 2023PEA3 transcription factor has been identified as a downstream target of the MAPK and PI3K pathways, and PEA3 overexpression has been observed in a variety of tumor...
BACKGROUND
PEA3 transcription factor has been identified as a downstream target of the MAPK and PI3K pathways, and PEA3 overexpression has been observed in a variety of tumor types. We aimed to evaluate PEA3 expression in odontogenic cysts and tumors and compare the expression among odontogenic lesions. In addition, the correlations between PEA3 expression and clinicopathological characteristics of conventional ameloblastoma and unicystic ameloblastoma were investigated.
METHODS
This study was performed on 165 samples of odontogenic cysts and tumors including 20 dentigerous cysts, 20 odontogenic keratocysts, 16 adenomatoid odontogenic tumors, 5 ameloblastic fibromas, 45 unicystic ameloblastomas, and 59 conventional ameloblastomas. The sections were immunohistochemically stained with mouse monoclonal anti-PEA3 antibody and PEA3 expression was evaluated as the immunoreactive score.
RESULTS
PEA3 expression was absent in all dentigerous cysts (DCs) and odontogenic keratocysts, while all adenomatoid odontogenic tumors showed either no (75%) or low (25%) expression of PEA3. Most of the ameloblastic fibromas (60%) displayed no PEA3 expression. A high expression of PEA3 was observed in a substantial number of unicystic ameloblastomas (48.9%) and conventional ameloblastomas (49.2%) in our study. PEA3 expression in DCs, odontogenic keratocysts and adenomatoid odontogenic tumors were significantly different from that in conventional ameloblastomas and that in unicystic ameloblastomas (p < 0.05). The expression of PEA3 was significantly different in the age groups of unicystic ameloblastomas and histological subtypes of conventional ameloblastomas (p < 0.05).
CONCLUSION
PEA3 overexpression is predominant in unicystic ameloblastomas and conventional ameloblastomas compared to other odontogenic lesions, indicating a pivotal role of PEA3 as a downstream effector of MAPK pathway in these two odontogenic lesions.
Topics: Ameloblastoma; Dentigerous Cyst; Fibroma; Jaw Neoplasms; Odontogenic Cysts; Odontogenic Tumors; Phosphatidylinositol 3-Kinases; Humans
PubMed: 37549030
DOI: 10.1111/jop.13476 -
Frontiers in Immunology 2023Ameloblastoma is a locally invasive and aggressive epithelial odontogenic neoplasm. The BRAF-V600E gene mutation is a prevalent genetic alteration found in this tumor...
BACKGROUND
Ameloblastoma is a locally invasive and aggressive epithelial odontogenic neoplasm. The BRAF-V600E gene mutation is a prevalent genetic alteration found in this tumor and is considered to have a crucial role in its pathogenesis. The objective of this study is to develop and validate a radiomics-based machine learning method for the identification of BRAF-V600E gene mutations in ameloblastoma patients.
METHODS
In this retrospective study, data from 103 patients diagnosed with ameloblastoma who underwent BRAF-V600E mutation testing were collected. Of these patients, 72 were included in the training cohort, while 31 were included in the validation cohort. To address class imbalance, synthetic minority over-sampling technique (SMOTE) is applied in our study. Radiomics features were extracted from preprocessed CT images, and the most relevant features, including both radiomics and clinical data, were selected for analysis. Machine learning methods were utilized to construct models. The performance of these models in distinguishing between patients with and without BRAF-V600E gene mutations was evaluated using the receiver operating characteristic (ROC) curve.
RESULTS
When the analysis was based on radiomics signature, Random Forest performed better than the others, with the area under the ROC curve (AUC) of 0.87 (95%CI, 0.68-1.00). The performance of XGBoost model is slightly lower than that of Random Forest, and its AUC is 0.83 (95% CI, 0.60-1.00). The nomogram evident that among younger women, the affected region primarily lies within the mandible, and patients with larger tumor diameters exhibit a heightened risk. Additionally, patients with higher radiomics signature scores are more susceptible to the BRAF-V600E gene mutations.
CONCLUSIONS
Our study presents a comprehensive radiomics-based machine learning model using five different methods to accurately detect BRAF-V600E gene mutations in patients diagnosed with ameloblastoma. The Random Forest model's high predictive performance, with AUC of 0.87, demonstrates its potential for facilitating a convenient and cost-effective way of identifying patients with the mutation without the need for invasive tumor sampling for molecular testing. This non-invasive approach has the potential to guide preoperative or postoperative drug treatment for affected individuals, thereby improving outcomes.
Topics: Humans; Female; Ameloblastoma; Proto-Oncogene Proteins B-raf; Retrospective Studies; Machine Learning; Mutation
PubMed: 37646022
DOI: 10.3389/fimmu.2023.1180908 -
Dental Clinics of North America Apr 2024This article addresses jaw lesions including cysts and benign odontogenic tumors in terms of their definition and clinical and imaging features and discusses pertinent... (Review)
Review
This article addresses jaw lesions including cysts and benign odontogenic tumors in terms of their definition and clinical and imaging features and discusses pertinent differential diagnoses..
Topics: Humans; Cysts; Jaw; Odontogenic Tumors; Diagnosis, Differential; Odontogenic Cysts
PubMed: 38417991
DOI: 10.1016/j.cden.2023.09.004