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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 -
Medicina Oral, Patologia Oral Y Cirugia... Mar 2021Desmoplastic Ameloblastoma (DA) is a rare, true neoplasm of jaws with reported incidence of 4-13% among other variants of Ameloblastoma, however this appears distinct... (Review)
Review
BACKGROUND
Desmoplastic Ameloblastoma (DA) is a rare, true neoplasm of jaws with reported incidence of 4-13% among other variants of Ameloblastoma, however this appears distinct than the classic Ameloblastoma in anatomical distribution and clinical presentation. This is often mistaken as a fibro-osseous lesion because of its similar radiological appearance.
MATERIAL AND METHODS
To describe the clinical, radiographic and histopathological characteristics through a series of new cases of histologically proven DA including a case of an exceptionally large, recurrent lesion along with retrospective analysis of cases from literature available for an improved understanding of the behaviour and prognosis of DA. A total of 50 cases were analysed for the anatomical distribution, radiographic presentation and management. Out of the 50 cases, 47 cases were from the English literature reported from 2011 to 2019 and 3 were new cases.
RESULTS
DA showed a slight male predilection (male: female=1.17:1) with a predominance in the fourth and fifth decade of life. Mandibular involvement (52%) was more commonly seen with a marked tendency for the anterior region. Radiographically, most of the lesions presented mixed radiopacity with radiolucency(80%) and root displacement was observed in only 70.27 % cases. Recurrence rate of 26 .47 % was observed. Cases treated with resection resulted in lesser recurrence as compared to those treated with enucleation and curettage.
CONCLUSIONS
DA is distinguished by a peculiar display of clinicalopathological parameters. DA has tendency of local disposition and propensity of recurrence, which thus necessitates its aggressive management. It is not possible to conclude or report on the aggressive/recurrent nature and appropriate treatment modality for DA due to inadequate follow-up results.
Topics: Ameloblastoma; Female; Humans; Male; Mandible; Mandibular Neoplasms; Neoplasm Recurrence, Local; Radiography; Retrospective Studies
PubMed: 33037797
DOI: 10.4317/medoral.24152 -
Cancer Management and Research 2021Ameloblastoma is a benign odontogenic neoplasm with a high local recurrence rate if the operation is not thorough. However, a useful clinical tool for the quantitative...
PURPOSE
Ameloblastoma is a benign odontogenic neoplasm with a high local recurrence rate if the operation is not thorough. However, a useful clinical tool for the quantitative assessment of the prognosis and risk of postoperative recurrence of ameloblastoma has not yet been constructed. This study aims to develop a prognostic nomogram model for ameloblastoma of the jaw to assist surgeons in surgical decision-making.
PATIENTS AND METHODS
Patients who underwent initial surgery for ameloblastoma in our department from October 2004 to March 2020 were enrolled and randomly divided into training and validation sets. Univariate and multivariate Cox proportional hazards regression analyses were performed to identify the independent prognostic factors, from which a nomogram for predicting 3-, 5- and 10-year recurrence-free survival (RFS) of ameloblastoma was constructed using the training set and internally validated using the validation set. The model performance was assessed by Harrell's concordance index (C-index) and calibration curves.
RESULTS
A total of 302 eligible patients with ameloblastoma were enrolled, 54 of whom were confirmed to relapse during the follow-up period of 6 to 191 months. Four independent predictors, including cortical bone perforation, root(s) resorption, WHO classification, and treatment pattern, were identified and included in the construction of a nomogram for recurrence-free survival (RFS), which showed promising calibration performance and discrimination in the training set (C-index 0.790, 95% confidence interval [CI] 0.735-0.845) and the validation set (C-index 0.734, 95% CI 0.599-0.869).
CONCLUSION
A favorable nomogram was developed that accurately predicted the RFS of patients with ameloblastoma based on individual characteristics. Risk stratification using the nomogram could optimize tailored therapy and follow-up.
PubMed: 34103995
DOI: 10.2147/CMAR.S307517 -
Indian Journal of Otolaryngology and... Oct 2022Ameloblastoma is a locally aggressive benign neoplasm of jaw that has high propensity to recur. Pathological subtype and intent of surgical excision during primary...
Ameloblastoma is a locally aggressive benign neoplasm of jaw that has high propensity to recur. Pathological subtype and intent of surgical excision during primary surgery are two factors that are known to affect the prognosis in these cases. Nevertheless, there are hardly any studies which have studied recurrent ameloblastoma cases. Apart from sharing a tertiary care cancer center's experience in managing a series of recurrent ameloblastoma cases, we discuss some of the recent literature related to pathophysiology and principles related to their surgical management. Retrospective chart review of all those recurrent ameloblastoma cases which were operated between October 2013 and January 2016 and were subsequently followed up for minimum of 3 years. Total of 9 recurrent ameloblastoma cases were operated in the study period. All our patients had less-radical or non-curative intent surgical treatment in the past, and current treatment consisted wide excision of the involved segment of mandible with free fibular reconstruction. Among the 6 patients who were followed up for more than 3 years, none developed recurrence at 56.5 months of mean follow up duration. Almost all these patients had optimal results with good quality of life in terms of oral speech intellectuality and cosmetic perception of self. Complete excision of the involved segment of bone with reconstruction using the composite free fibular graft can provide optimal functional outcomes and can significantly lessen the chances of further recurrences.
PubMed: 36452534
DOI: 10.1007/s12070-020-01790-4 -
Journal of Oral and Maxillofacial... 2024Glucose uptake may be considered the rate-limiting step for the growth and metabolism of the cancer cell. Studies on GLUT1 have shown that GLUT1 is involved in cell...
CONTEXT
Glucose uptake may be considered the rate-limiting step for the growth and metabolism of the cancer cell. Studies on GLUT1 have shown that GLUT1 is involved in cell survival and proliferation in both healthy and pathological circumstances. GLUT1 expression is regarded as one of the crucial elements in the development of local aggressiveness, tumour invasiveness, and metastasis, particularly in malignant tumours. The role of glut1 in odontogenic cysts and tumours has remained uncertain.
AIM
The aim of the study is to assess the expression of Glut1 in dentigerous cysts, odontogenic keratocysts, and ameloblastoma.
SETTINGS AND DESIGN
The study was conducted in GSL Dental College. The study design was a resprospective immunohistochemical study.
METHODS AND MATERIAL
Formalin-fixed, paraffin-embedded blocks of histologically confirmed cases (n = 50), 10 cases of odontogenic keratocysts, dentigerous cysts, ameloblastomas solid, ameloblastomas unicystic, and dental follicles each. Brown colour staining was considered as positive staining for GLUT1. Quantitative analysis was performed by counting the number of labelled cells, and semi-quantitative analysis was conducted by assigning immunostaining intensity scores.
STATISTICAL ANALYSIS
Chi-square test was used to compare differences between the groups. A value of ≤0.05 was considered as statistically significant.
RESULTS
Odontogenic keratocysts and unicystic ameloblastoma showed ≥50% of label cells with strong intensity of staining. Odontogenic keratocysts and solid ameloblastoma showed sub-cellular localisation of staining in the cytoplasm and membrane. Dentigerous cysts exhibited combined nucleus, cytoplasm, and membrane sub-cellular localisation of staining.
CONCLUSIONS
The development of ameloblastomas, odontogenic keratocysts, and dentigerous cysts appears to be influenced by GLUT-1. Variation in its expression may aid in explanation of some of the differences in biological activity of these lesions.
PubMed: 38800443
DOI: 10.4103/jomfp.jomfp_455_23 -
International Journal of Stem Cells Nov 2022Cancer initiation and progression are profoundly along with the crosstalk between cancer cells and the surrounding stroma. Accumulating evidence has shown that the...
Cancer initiation and progression are profoundly along with the crosstalk between cancer cells and the surrounding stroma. Accumulating evidence has shown that the therapy targeting the extracellular matrix (ECM) would regress tumor growth and invasion in the most common carcinomas. However, it remains largely unexplored in several rare tumors like odontogenic tumors. Ameloblastoma (AM) is the representative odontogenic epithelial tumor in the jawbone, and it usually infiltrates into adjacent bone marrow and has unlimited growth capacity and a high potential for recurrence. This study aims to investigate the role of collagen-rich ECM during the invasion of AM. Transcriptomic analysis revealed that ECM- and epithelial-to-mesenchymal transition (EMT)-related genes were up-regulated in AM compared to ameloblastoma cell line, AM-1. Tumoroid forming analysis showed that Collagen-rich ECM is indispensable for AM progression, especially for aggressive growth patterns and collective invasion.
PubMed: 36310025
DOI: 10.15283/ijsc22132 -
Frontiers in Oncology 2022FOSL1, a key component of the Activating protein-1 (AP-1) transcriptional complex, plays an important role in cancer cell migration, invasion, and proliferation....
BACKGROUND
FOSL1, a key component of the Activating protein-1 (AP-1) transcriptional complex, plays an important role in cancer cell migration, invasion, and proliferation. However, the impact of FOSL1 in ameloblastoma (AM) has not been clarified. Herein, we aimed to assess the expression of FOSL1 and investigate its functional role in AM.
METHODS
The expression of FOSL1 was examined based on an immunohistochemistry analysis of 96 AM samples. Cell proliferation, migration, invasion, and tumorigenesis were assessed using Cell Counting Kit-8 (CCK-8), colony formation, Transwell, and sphere formation assays. RNA sequencing (RNA-seq) was employed to investigate the molecular alterations of AM cells upon FOSL depletion. Microarrays of AMs were downloaded from the Gene Expression Omnibus (GEO) database for bioinformatics analysis. In addition, patient-derived AM organoids were used to evaluate the therapeutic value of the AP-1 inhibitor.
RESULTS
FOSL1 was detected in the nuclei of AMs and upregulated in conventional AMs compared to unicystic AMs and normal oral epithelium. Compared with primary AM, FOSL1 expression was significantly increased in recurrent AM. Genetic knockdown of FOSL1 suppressed the proliferation, migration, invasion, and sphere formation of AMs. Similar results were also observed by pharmacological inhibition of AP-1 activity. Moreover, the AP-1 inhibitor T5224 impeded the growth of organoids derived from AM patients. Mechanistically, our Ingenuity Pathway Analysis (IPA) and gene set enrichment analysis (GSEA) results revealed that depletion of FOSL1 inactivated kinetochore metaphase signaling and the epithelial-mesenchymal transition pathway and then impaired the aggressiveness of AM cells accordingly.
CONCLUSION
FOSL1 promotes tumor recurrence and invasive growth in AM by modulating kinetochore metaphase signaling and the epithelial-mesenchymal transition pathway; thus, it represents a promising therapeutic target for AM treatment.
PubMed: 36185257
DOI: 10.3389/fonc.2022.900108 -
BMJ Case Reports Dec 2021Ameloblastic carcinoma is a rare malignant odontogenic neoplasm that exhibits diverse clinical and radiological presentations. In fact there are several differential...
Ameloblastic carcinoma is a rare malignant odontogenic neoplasm that exhibits diverse clinical and radiological presentations. In fact there are several differential diagnoses during histopathological evaluation too. Lack of adequate reports could not establish the predominant demographic, clinical and radiological presentations. For the same reasons, the role of adjuvant radiotherapy and chemotherapy is also unsubstantiated yet. This case discusses the innocuous clinical and radiological presentation of ameloblastic carcinoma in a 55-year-old man where the diagnostic confirmation was achieved through histopathological evaluation. The differential diagnoses, treatment and follow-up details of this case are discussed in light of the previous published case reports and systematic reviews of case reports in an attempt to increase the sensitisation among dentists towards ameloblastic carcinoma.
Topics: Ameloblastoma; Carcinoma; Diagnosis, Differential; Humans; Male; Mandibular Neoplasms; Middle Aged; Odontogenic Tumors
PubMed: 34906959
DOI: 10.1136/bcr-2021-246907 -
Cureus Oct 2023Ameloblastoma is one of the most prevalent but enigmatic benign odontogenic tumors of the jaw, accounting for approximately 10% of all maxillary and mandibular tumors.... (Review)
Review
Ameloblastoma is one of the most prevalent but enigmatic benign odontogenic tumors of the jaw, accounting for approximately 10% of all maxillary and mandibular tumors. This neoplasia is distinguished by exhibiting several clinical and histological variants along with several mutations that affect its behavior. The ameloblastoma treatment plan is determined by the tumor's size, anatomical location, histologic variant, and anatomical involvement. On chromosome 7, there is a proto-oncogene called BRAF. When BRAF is mutated, it becomes an oncogene and continuously produces proteins like MEK and ERK, members of mitogen-activated protein kinase (MAPK). In the signaling pathway, these proteins activate transcription factor inside the nucleus that helps in cell division and growth. Numerous neoplasms have been linked to more than 40 BRAF mutations. The most common one is BRAF proto-oncogene serine/threonine kinase (BRAF) V600E, whose treatment has been linked to a positive outcome. BRAF inhibitors like vemurafenib, dabrafenib, and sorafenib have shown excellent results, especially in metastatic ameloblastoma. BRAF, particularly in the case of metastatic ameloblastoma, inhibitors such as vemurafenib, dabrafenib, and sorafenib, has demonstrated outstanding results. Targeted therapies have been employed as adjuvant therapies to enhance cosmetic outcomes, even though no reports of serial cases demonstrate their effectiveness in ameloblastomas. In the treatment of ameloblastomas, the identification of BRAF V600E and additional mutations as the prime targeted therapies has proven to be a significant breakthrough where surgical treatment was contraindicated. In this article, we review the presence of BRAF V600E mutations, their inhibitors, and targeted therapies in ameloblastoma.
PubMed: 38021761
DOI: 10.7759/cureus.47682 -
Medicina Oral, Patologia Oral Y Cirugia... Jul 2022Ameloblastomas are benign odontogenic tumors that can eventually mimic the clinical and radiological features of apical periodontitis. The aim of the present study was...
BACKGROUND
Ameloblastomas are benign odontogenic tumors that can eventually mimic the clinical and radiological features of apical periodontitis. The aim of the present study was to evaluate the clinical, radiological and histological characteristics from a series of ameloblastomas mimicking apical periodontitis diagnosed in a 14-year period.
MATERIAL AND METHODS
all cases histologically diagnosed as ameloblastomas from 2005 to 2018 presenting a clinical diagnosis of periapical lesion of endodontic origin were selected for the study. Clinical, radiological and histological characteristics from all cases were tabulated and descriptively and comparatively analyzed.
RESULTS
Twenty cases composed the final sample, including 18 solid and 2 unicystic ameloblastomas. Mean age of the affected patients was in the fifth decade with predilection for males (72%). The most common anatomical location was the posterior mandible (55%) and most cases presented a radiolucent unilocular (80%) well-defined (95%) image. Most cases were asymptomatic, but the presence of local swelling and bone cortical rupture were common.
CONCLUSIONS
Ameloblastomas mimicking periapical lesions of endodontic origin are mostly diagnosed in adult males as well-defined radiolucent unilocular lesions producing local swelling and bone cortical rupture.
Topics: Adult; Ameloblastoma; Humans; Male; Odontogenic Tumors; Periapical Periodontitis; Radiography
PubMed: 35660730
DOI: 10.4317/medoral.25338