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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 -
Journal of Comparative Pathology Aug 2022A 2-year-old male Thoroughbred horse presented with a mass in the maxilla. The focally ulcerated mass, approximately 8 cm in diameter, covered the upper left...
A 2-year-old male Thoroughbred horse presented with a mass in the maxilla. The focally ulcerated mass, approximately 8 cm in diameter, covered the upper left intermediate and corner incisor teeth (nos. 602 and 603 according to the modified Triadan system) and radiographic examination revealed displacement and lysis of the incisors. Histologically, the tumour was composed of a dense proliferation of spindle-shaped cells and neoplastic odontogenic epithelial cells arranged in island, follicular, plexiform or sheetlike patterns. The spindle-shaped cells were immunopositive for cytokeratins AE1/AE3, 5/6, 14 and 19. The Ki-67 index was 32.6% in the spindle cell component. Based on the histological and immunohistochemical findings, the tumour was diagnosed as spindle cell ameloblastic carcinoma.
Topics: Ameloblastoma; Animals; Carcinoma; Horse Diseases; Horses; Male
PubMed: 36008038
DOI: 10.1016/j.jcpa.2022.05.006 -
Medicina Oral, Patologia Oral Y Cirugia... Sep 2022Ep-CAM, a transmembrane glycoprotein expressed in most epithelium in normal conditions, has diverse roles in these tissues, including in cell adhesion, proliferation,...
BACKGROUND
Ep-CAM, a transmembrane glycoprotein expressed in most epithelium in normal conditions, has diverse roles in these tissues, including in cell adhesion, proliferation, differentiation, cell cycle regulation, migration and intracellular signaling. It is also over-expressed in most malignant neoplasia, participating in the initiation, progression, and metastatic dissemination of the tumor. The expression and roles of this protein in oral neoplasia, particularly in odontogenic tumors, remain unestablished. The objective of this study consisted in analyzing the expression of this protein in ameloblastoma and tooth germ.
MATERIAL AND METHODS
Ep-CAM (MOC-31) expression was evaluated by immunohistochemistry in tooth germs (TG) (n = 16) ameloblastomas (AM) (n = 60) and 2 ameloblastic carcinomas. Sections were visualized in their totality with an optical microscope, and positivity observed in cell membrane and cytoplasm was graded according to the following semi-quantitative scale: Neg, "essentially unstained", for negative sections or staining <5% of cells; + for staining of 5-50% of cells; ++ for staining >50% of cells.
RESULTS
Most tooth germs expressed MOC-31 (81.3%), strong staining was observed both in the inner epithelium of the enamel organ and in the adjacent stellate reticulum. 16.7% of the AM cases showed MOC-31 expression, the immunoexpression expression was diffuse at the cytoplasmic and membrane level. The only two cases of ameloblastic carcinoma included were strong positive to MOC-31. No correlation was observed between protein expression and gender, age, clinical variants, or histological subtypes.
CONCLUSIONS
Overexpression was found in TG and ameloblastic carcinoma compared to AM; further studies with different experimental strategies are suggested to clarify the biological significance of this finding.
Topics: Ameloblastoma; Carcinoma; Epithelial Cell Adhesion Molecule; Humans; Odontogenic Tumors; Tooth Germ
PubMed: 35975801
DOI: 10.4317/medoral.25145 -
Journal of Oral Pathology & Medicine :... Aug 2022To investigate and compare the demographic data, occurrence of recurrence and metastasis, and survival prognosis between ameloblastic carcinoma (AC) and metastasizing... (Review)
Review
PURPOSE
To investigate and compare the demographic data, occurrence of recurrence and metastasis, and survival prognosis between ameloblastic carcinoma (AC) and metastasizing ameloblastoma (MA), based on appropriate and currently accepted eligible diagnostic criteria, in a systematic review of the literature.
METHODS
An electronic search was undertaken, last updated in December 2021. Eligibility criteria included publications having enough clinicopathological information to confirm the diagnosis of these tumors.
RESULTS
Seventy-seven publications reporting 85 ACs and 43 MAs were included. Both tumors were more frequent in mandible and showed different clinical profiles regarding patients' sex and age. There was no difference in the estimated cumulative survival between patients diagnosed with these tumors. Metastases mainly affected the lungs, followed by cervical lymph nodes. The mean time between the first metastasis and the last follow-up was higher for MA (p = 0.021). In addition, MA patients remained alive longer than AC patients after the first metastasis diagnosis (p = 0.041). Considering only the cases that metastasized, a higher ratio of AC patients died in comparison to MA patients (p = 0.003). The occurrence of recurrence was associated with a conservative primary treatment with both AC (p < 0.001) and MA tumors (p = 0.017). Multiple recurrent events were associated with conservative primary therapies with MA (p < 0.001) but not with AC (p = 0.121).
CONCLUSION
In addition to some demographic differences, ACs that metastasize present a worse prognosis than MA. As conservative procedures are associated with multiple recurrent events, this treatment modality should be avoided for both tumors.
Topics: Ameloblastoma; Carcinoma; Humans; Mandible; Mandibular Neoplasms; Odontogenic Tumors
PubMed: 35822408
DOI: 10.1111/jop.13334 -
Oral Oncology Sep 2022
Topics: Carbazoles; Carcinoma; Humans; Lung Neoplasms; Piperidines; Protein Kinase Inhibitors; Receptor Protein-Tyrosine Kinases
PubMed: 35759857
DOI: 10.1016/j.oraloncology.2022.105946 -
International Journal of Surgical... Jun 2023Ameloblastic carcinoma is a rare malignant neoplasm arising from the odontogenic epithelium. Ameloblastic carcinoma commonly occurs de novo affecting the posterior... (Review)
Review
Ameloblastic carcinoma is a rare malignant neoplasm arising from the odontogenic epithelium. Ameloblastic carcinoma commonly occurs de novo affecting the posterior segments of the mandible. Presently, only less than 100 cases have been reported arising from the maxilla. We report a unique case of maxillary ameloblastic carcinoma in a 68-year-old male with a 5.6 cm positron emission tomography (PET) avid left maxillary sinus mass. The patient underwent a left maxillectomy which revealed hyperchromatic and pleomorphic tumor cells arranged in a nested and trabecular architecture. The tumor cells showed distinct peripheral palisading with reverse polarization. Areas of bone destruction, necrosis, lymphovascular and perineural invasions, as well as atypical mitoses, were identified. Immunohistochemically, the tumor cells were positive for keratin cocktail (AE1/AE3 and CAM 5.2), keratin 19, p40, and weakly positive for MDM2, while negative for calretinin. Molecular analysis revealed wild-type ; however, alterations in /, , , , , , , and were present. This histopathologic and molecular profile supported the diagnosis of ameloblastic carcinoma. There has been no evidence of disease recurrence or metastasis eleven months after the initial diagnosis.
Topics: Male; Humans; Aged; Maxilla; Odontogenic Tumors; Mandible; Carcinoma; Ameloblastoma; DNA Helicases; Nuclear Proteins; Transcription Factors; Fibroblast Growth Factors
PubMed: 35668625
DOI: 10.1177/10668969221102542 -
Journal of Oral and Maxillofacial... Feb 2022Malignant odontogenic neoplasms are extremely challenging to study due to their rarity and variable clinical presentations. Ameloblastic carcinoma (AC) is one such...
Malignant odontogenic neoplasms are extremely challenging to study due to their rarity and variable clinical presentations. Ameloblastic carcinoma (AC) is one such odontogenic tumor which has been the subject of controversy, in part because of its scarcity, complicated by confusion in terminology along with complexity in classification. Histologic features of AC resemble tumor cells of ameloblastoma but exhibit cellular atypia. Surgical resection for this kind of lesion, leaving at least a 2 cm free margin coupled with neoadjuvant radiotherapy, might prove fruitful results. The current paper reports a case of an extraosseous variant of AC which posed a diagnostic challenge due to variable presentations histopathologically, suggesting the need for evidence-based case studies and molecular workup for a better therapeutic and prognostic insight.
PubMed: 35450254
DOI: 10.4103/jomfp.jomfp_378_21 -
Annals of Translational Medicine Dec 2021Ameloblastic carcinoma (AC) is an extremely rare malignant odontogenic tumor. The mean age of occurrence for all 141 AC cases analyzed in our systematic review study was...
Ameloblastic carcinoma (AC) is an extremely rare malignant odontogenic tumor. The mean age of occurrence for all 141 AC cases analyzed in our systematic review study was 43.59±19.51 years. Males were more affected than females, and the mandible was predominantly affected compared with the maxilla. The main clinical manifestation was a painful or painless swelling with ulceration and radiographic features usually displayed as mixed cystic or solid changes. Surgical resection was the first recommended method of management. Fourteen cases had cervical lymph node spread, 19 had distant metastasis (most commonly in the lung), and 33 had recurrence. We present a rare case of AC involving the maxillary region. Locally extensive surgical resection was carried out. Ablative defects after maxillectomy resulted in the communication of oral cavity and nasal cavity/maxillary antrum and would bring about difficulties in mastication, deglutition, and speech. A submental island flap was applied to close the oronasal and oroantral fistula. The flap and the wounds healed well, with excellent outcomes in terms of appearance, the function of speech, and swallowing on follow up. The submental island flap provides a relatively thin, easy-to-harvest, and well-vascularized tissue, which makes it a reliable option in soft tissue reconstruction of the oral and maxillofacial region.
PubMed: 35071440
DOI: 10.21037/atm-21-5196 -
Journal of Personalized Medicine Jan 2022Ameloblastoma is the most common benign odontogenic neoplasm, but with an aggressive behavior and a high recurrence rate. Nowadays wide surgical resection is the current...
Ameloblastoma is the most common benign odontogenic neoplasm, but with an aggressive behavior and a high recurrence rate. Nowadays wide surgical resection is the current recommended treatment, which can cause further loss of function and esthetics. Recent studies point to the stem/progenitor cells as both initiators and propagators of the tumors. Elucidation of the cellular and molecular mechanisms underlying the tumor stem cells is of broad interest for understanding tumorigenesis and for developing effective targeted therapies. SRY related HMG box gene 2 (SOX2) is a transcription factor that plays important roles in development, stem cell renewal, and cancer formation. Few studies have revealed increased SOX2 expression in atypical ameloblastoma and ameloblastic carcinoma. For the development of personalized medicine for ameloblastoma, biomarkers that provide prognostic or predictive information regarding a tumor's nature or its response to treatment are essential. Thus, in this study, we aimed to study if SOX2-positive cells exist in ameloblastomas and their correlation with the clinicopathologic parameters. Our data suggested BRAF(V600E) mutation might contribute to the expansion of SOX2-positive cells. The identification of BRAF(V600E) mutation and the amplification of SOX2-positive cells in ameloblastomas imply the possible benefit of applying BRAF and SOX2 inhibitors in recurrent and un-resectable ameloblastomas.
PubMed: 35055392
DOI: 10.3390/jpm12010077 -
Frontiers in Oral Health 2021Malignant odontogenic tumours (MOTs) arise either from the tooth forming tissues, their developmental residues or from existing odontogenic epithelial or mesenchymal...
Malignant odontogenic tumours (MOTs) arise either from the tooth forming tissues, their developmental residues or from existing odontogenic epithelial or mesenchymal neoplasms in the jaws. Their management requires extensive surgery due to their infiltrative nature and risk of metastasis. There is a need to understand the clinical and pathological features of MOTs to inform both treatment algorithms and prognostication. This is an area of diagnostic pathology which presents substantial difficulties in diagnosis, compounded by inconsistent use of terminology. Thus, this systematic review aimed to describe the clinical and pathological features of MOTs with a view to consolidating the literature and defining problematic areas in diagnosis and classification. An electronic database search was conducted in Web of Science, PubMed/Medline, and Embase. Additionally, the grey literature and reference lists of selected papers searched for completeness. Nine hundred and sixty articles were initially identified. Following removal of duplicates and application of inclusion/exclusion criteria, 312 articles were included for qualitative analysis. The 312 articles encompassed a total of 507 patients with most lesions located within the mandible (74.3%). The most common first histological diagnosis was ameloblastic carcinoma (25.7% of all diagnoses), but there is considerable variation in how and when various diagnostic terms are used, and several misdiagnoses were reported. An initial benign diagnosis was made in 24.7% of patients, followed by a later malignant diagnosis and in this sub-group, the most common benign first diagnosis was ameloblastoma (42.4%). Cervical lymph nodes were the most common site of metastasis (9.3% of patients). With respect to distant metastasis (DM), the lungs were the most common organ affected (11.2% of DM patients) with metastasising ameloblastoma the most commonly reported tumour which metastasised to the lungs. Overall, 26.8% of patients developed recurrence. Overall, the quality of the literature on MOTs is poor. This review of the literature has highlighted variations in diagnostic terms and criteria which has resulted in areas of confusion with potential for misdiagnosis. This consolidation of primary data has identified key areas for targeted research including further discussion on the malignant potential of ameloblastoma.
PubMed: 35048074
DOI: 10.3389/froh.2021.775707