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Journal of Oral Pathology & Medicine :... Nov 2023Odontogenic tumors (OT) are composed of heterogeneous lesions, which can be benign or malignant, with different behavior and histology. Within this classification,...
BACKGROUND
Odontogenic tumors (OT) are composed of heterogeneous lesions, which can be benign or malignant, with different behavior and histology. Within this classification, ameloblastoma and ameloblastic carcinoma (AC) represent a diagnostic challenge in daily histopathological practice due to their similar characteristics and the limitations that incisional biopsies represent. From these premises, we wanted to test the usefulness of models based on artificial intelligence (AI) in the field of oral and maxillofacial pathology for differential diagnosis. The main advantages of integrating Machine Learning (ML) with microscopic and radiographic imaging is the ability to significantly reduce intra-and inter observer variability and improve diagnostic objectivity and reproducibility.
METHODS
Thirty Digitized slides were collected from different diagnostic centers of oral pathology in Brazil. After performing manual annotation in the region of interest, the images were segmented and fragmented into small patches. In the supervised learning methodology for image classification, three models (ResNet50, DenseNet, and VGG16) were focus of investigation to provide the probability of an image being classified as class0 (i.e., ameloblastoma) or class1 (i.e., Ameloblastic carcinoma).
RESULTS
The training and validation metrics did not show convergence, characterizing overfitting. However, the test results were satisfactory, with an average for ResNet50 of 0.75, 0.71, 0.84, 0.65, and 0.77 for accuracy, precision, sensitivity, specificity, and F1-score, respectively.
CONCLUSIONS
The models demonstrated a strong potential of learning, but lack of generalization ability. The models learn fast, reaching a training accuracy of 98%. The evaluation process showed instability in validation; however, acceptable performance in the testing process, which may be due to the small data set. This first investigation opens an opportunity for expanding collaboration to incorporate more complementary data; as well as, developing and evaluating new alternative models.
Topics: Humans; Ameloblastoma; Artificial Intelligence; Deep Learning; Reproducibility of Results; Odontogenic Tumors; Carcinoma
PubMed: 37712132
DOI: 10.1111/jop.13481 -
Indian Journal of Otolaryngology and... Sep 2023Ameloblastic carcinoma is an odontogenic neoplasm with combined features of ameloblastoma and carcinoma on histopathological examination. Its prognosis is dominated by...
Ameloblastic carcinoma is an odontogenic neoplasm with combined features of ameloblastoma and carcinoma on histopathological examination. Its prognosis is dominated by risk of local recurrence and distant metastasis. We report our patient because of the rare site and to highlight the importance of early, aggressive surgical treatment and regular follow-up.
PubMed: 37636673
DOI: 10.1007/s12070-023-03758-6 -
BMC Oral Health Aug 2023Ameloblastic carcinoma and metastasising ameloblastoma are rare epithelial odontogenic tumours with aggressive features. Distinguishing between these two lesions is... (Review)
Review
BACKGROUND
Ameloblastic carcinoma and metastasising ameloblastoma are rare epithelial odontogenic tumours with aggressive features. Distinguishing between these two lesions is often clinically difficult but necessary to predict tumour behaviour or to plan future therapy. Here, we provide a brief review of the literature available on these two types of lesions and present a new case report of a young man with an ameloblastoma displaying metastatic features. We also use this case to illustrate the similarities and differences between these two types of tumours and the difficulties of their differential diagnosis.
CASE PRESENTATION
Our histopathological analyses uncovered a metastasising tumour with features of ameloblastic carcinoma, which developed from the ameloblastoma. We profiled the gene expression of Wnt pathway members in ameloblastoma sample of this patient, because multiple molecules of this pathway are involved in the establishing of cell polarity, cell migration or for epithelial-mesenchymal transition during tumour metastasis to evaluate features of tumor behaviour. Indeed, we found upregulation of several cell migration-related genes in our patient. Moreover, we uncovered somatic mutation BRAF p.V600E with known pathological role in cancerogenesis and germline heterozygous FANCA p.S858R mutation, whose interpretation in this context has not been discussed yet.
CONCLUSIONS
In conclusion, we have uncovered a unique case of ameloblastic carcinoma associated with an alteration of Wnt signalling and the presence of BRAF mutation. Development of harmful state of our patient might be also supported by the germline mutation in one FANCA allele, however this has to be confirmed by further analyses.
Topics: Male; Humans; Ameloblastoma; Proto-Oncogene Proteins B-raf; Odontogenic Tumors; Mutation; Carcinoma
PubMed: 37573343
DOI: 10.1186/s12903-023-03259-6 -
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 -
Pathology International Aug 2023Ghost cell odontogenic carcinoma (GCOC) is an extremely rare intraosseous malignant odontogenic tumor with prominent ghost cell keratinization and dentinoid formation....
Ghost cell odontogenic carcinoma (GCOC) is an extremely rare intraosseous malignant odontogenic tumor with prominent ghost cell keratinization and dentinoid formation. Here, we present the first case of GCOC arising in dentinogenic ghost cell tumor (DGCT), peripheral. The patient was a man in his 60s with an exophytic mass in the anterior part of lower gingiva. The resected tumor measured 4.5 cm in maximum diameter. Histologically, the nonencapsulated tumor proliferated in the gingiva without bone invasion. It was predominantly composed of ameloblastoma-like nests and islands of basaloid cells with ghost cells and dentinoid in the mature connective tissue, suggesting DGCT, peripheral. As minor components, sheets of atypical basaloid cells and ameloblastic carcinoma-like nests with pleomorphism and high proliferative activity (Ki-67 labeling index up to 40%) consistent with malignancy were identified. CTNNB1 mutation and β-catenin nuclear translocation were observed in both benign and malignant components. Final diagnosis was GCOC arising in DGCT, peripheral. GCOC shows similar histological features to DGCT. In this unique case without invasion, the cytological atypia and high proliferative activity supports the diagnosis of malignant transformation from DGCT.
Topics: Male; Humans; Odontogenic Tumors; Jaw Neoplasms; Ameloblastoma; Cell Transformation, Neoplastic; Carcinoma
PubMed: 37401869
DOI: 10.1111/pin.13351 -
Journal of Stomatology, Oral and... Dec 2023Clinico-histopathologic assessment of patients with ameloblastoma and ameloblastic carcinoma remains the best diagnostic modality for the tumors. However, in cases where...
BACKGROUND
Clinico-histopathologic assessment of patients with ameloblastoma and ameloblastic carcinoma remains the best diagnostic modality for the tumors. However, in cases where the criteria for arriving at a definitive diagnosis are not clearcut, the pathologist is faced with a dilemma and thus an imperative need for adjunct diagnostic methods.
OBJECTIVES
To evaluate/compare the immunohistochemical expression of NM23 in classical, borderline (atypical) ameloblastoma and ameloblastic carcinoma and to assess usefulness of NM23 in closing diagnostic gaps between ameloblastoma and ameloblastic carcinoma.
METHODS
Twenty-four (24) cases of ameloblastoma, 10 ameloblastoma with classical histopathologic features, 8 with nonclassical histopathology [atypical], and 6 cases of ameloblastic carcinoma were selected from cases seen at the Oral Pathology Laboratory of the Lagos State University College of Medicine, Nigeria. NM23 immunostaining protocol was done on the selected tissue blocks and evaluated using Sinicrope method. Analysis was done using R language.
RESULTS
Positive NM23 staining was observed in all cases of ameloblastoma and ameloblastic carcinoma, with more intense staining observed in the stellate reticulum-like areas than in the ameloblast-like areas. Ameloblastic carcinoma stained intensely with NM23 (100%) compared with atypical cases (37.5%) and ameloblastoma (20.0%; p = 0.04). The mean aggregate score was also significantly higher in AC (11 ± 2.4; p = 0.01). The mean aggregate score was also significant amongst growth pattern of ameloblastoma (p = 0 0.02).
CONCLUSIONS
The findings in this study reveal the usefulness of NM23 in differentiating ameloblastoma from ameloblastic carcinoma; a more comprehensive study with a larger sample size is recommended to corroborate or refute the findings in this study.
Topics: Humans; Ameloblastoma; Nigeria; Odontogenic Tumors; Cell Proliferation; Carcinoma
PubMed: 37295743
DOI: 10.1016/j.jormas.2023.101532 -
International Journal of Surgical... Aug 2023
Topics: Humans; Odontogenic Tumors; Ameloblastoma; Carcinoma; Mandibular Neoplasms
PubMed: 36071617
DOI: 10.1177/10668969221122993 -
International Journal of Surgical... Sep 2023Ameloblastic carcinoma (AC) is an uncommon malignant odontogenic tumor, with about 200 cases published in the literature. Here, we report a case of AC occurring in the...
Ameloblastic carcinoma (AC) is an uncommon malignant odontogenic tumor, with about 200 cases published in the literature. Here, we report a case of AC occurring in the maxilla - at the same anatomical location of an ameloblastoma 8 years earlier. A 23-year-old woman with maxillary swelling of 6-month duration was referred to our service. Extraoral examination revealed a swelling over the right side of the face, lifting her lip and nose. Intraorally, a firm, reddish, nodular swelling involving the right maxilla was observed. Computed tomography showed a hypodense image invading the right maxilla, maxillary sinus, and nasal fossa. An incisional biopsy was performed, followed by histopathologic examination and an immunohistochemical panel, which led to the diagnosis of AC. The lesion was treated with partial maxillectomy followed by immediate rehabilitation. Neither local recurrence of the tumor nor distant metastasis was observed during a 4-year follow-up. AC is a highly malignant lesion which requires aggressive therapy. Its diagnosis should be based on combined clinical, imaging, and pathological manifestations in order to improve diagnostic accuracy.
Topics: Female; Humans; Odontogenic Tumors; Ameloblastoma; Maxilla; Tomography, X-Ray Computed; Carcinoma
PubMed: 36031847
DOI: 10.1177/10668969221118323