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Medical Oncology (Northwood, London,... Apr 2023Ameloblastoma in 66% of the cases harbor a somatic mutation of the "mitogen-activated protein kinase" signaling pathway (BRAF V600E). In V600E mutations, BRAF is in the... (Review)
Review
BACKGROUND
Ameloblastoma in 66% of the cases harbor a somatic mutation of the "mitogen-activated protein kinase" signaling pathway (BRAF V600E). In V600E mutations, BRAF is in the permanent "on" state and relays the growth-promoting signals independently of the EGFR pathway. Therefore, mutant BRAF represents a target for handful of new drugs.
METHODS
We conducted a literature search, with the search terms "Vemurafenib, Dabrafenib, Ameloblastoma, and BRAF." These included seven case reports with nine patients who underwent monotherapy with Dabrafenib or Vemurafenib or combination therapy with Dabrafenib and Trametinib.
RESULTS
The patients age ranges from 10 years up to 86 years. The distribution of women and men is 4:5. Patients with an initial diagnosis of ameloblastoma, as well as recurrences or metastasized ameloblastoma were treated. Indications cover neoadjuvant therapy up to the use in metastasized patients in an irresectable state. Results ranging from "only" tumor size reduction to restitutio ad integrum.
CONCLUSION
We see the use of BRAF Inhibitors to reduce tumor size with consecutive surgical treatment as a reasonable option for therapy. However, we are aware that at present the data are based only on case reports with the longest follow-up of just 38 months. We encourage further clinical trials in the use of BRAF Inhibitors for selecting ameloblastoma patients in a multi-center setting.
Topics: Male; Humans; Female; Child; Vemurafenib; Proto-Oncogene Proteins B-raf; Ameloblastoma; Imidazoles; Protein Kinase Inhibitors; Mutation; Antineoplastic Combined Chemotherapy Protocols
PubMed: 37115331
DOI: 10.1007/s12032-023-01993-z -
The British Journal of Oral &... Dec 2021In this study, we aimed to systematically review and critically appraise the available literature concerning the effectiveness of marsupialisation and decompression on... (Review)
Review
In this study, we aimed to systematically review and critically appraise the available literature concerning the effectiveness of marsupialisation and decompression on the reduction of cystic jaw lesions. The 'Preferred Reporting Items for Systematic Reviews and Meta-Analysis' guidelines were followed and the study protocol was registered at the 'International Prospective Register of Systematic Reviews' (CRD42019116099). Six main databases were searched: Embase, LILACS, PubMed, Scopus, The Cochrane Library, and Web of Science. Searches were complemented with three grey literature sources: Google Scholar, ProQuest, and Open Grey. Any reduction measures, compared with preoperative status or other procedures, were considered. Risk of bias was assessed using the Joanna Briggs Institute Critical Appraisal Tool. Thirty-one studies were included, of which five were judged with low, 24 with moderate, and two with high risk of bias. Considering surgical approach, 20 studies assessed the decompression and 11 the marsupialisation technique. Most studies considered these techniques as preliminary treatments, followed by enucleation. From 1088 lesions found, most were odontogenic keratocysts (33.8%), followed by unicystic ameloblastomas (21.0%), dentigerous cysts (20.6%), and radicular cysts (8.4%). Large lesions and younger individuals frequently presented more favourable responses to treatment and anatomical location was not associated with lesion reduction overall. The intervention duration generally ranged between one to two years. In conclusion, marsupialisation and decompression were mostly considered as preliminary treatments, followed by enucleation. Lesion reduction was generally considered insufficient for these techniques to be used as definitive therapies, although benefits concerning the diminished invasiveness of the secondary surgery were often proposed.
Topics: Ameloblastoma; Decompression, Surgical; Humans; Odontogenic Cysts; Odontogenic Tumors
PubMed: 34749963
DOI: 10.1016/j.bjoms.2021.03.004 -
Journal of Oral Pathology & Medicine :... Nov 2023CTNNB1 gene encodes beta catenin, a transcriptional activator of Wnt pathway involved in the pathogenesis of odontogenic lesions. Though located intramembranously, its... (Review)
Review
BACKGROUND
CTNNB1 gene encodes beta catenin, a transcriptional activator of Wnt pathway involved in the pathogenesis of odontogenic lesions. Though located intramembranously, its translocation into cytoplasm and nucleus could trigger cell proliferation, inhibition of apoptosis, invasion and migration of the tumour cell.
MATERIALS AND METHODS
Five electronic databases including MEDLINE by PubMed, Google scholar, Scopus, Trip, Cochrane library and EMBASE until 1 January 2023 without period restriction were thoroughly searched. Those articles that identified CTNNB1 mutation and beta catenin in odontogenic lesions were included for review. Risk of bias was analysed for each study using QUADAS 2 tool and Review Manager 5.3 was used to output its result.
RESULTS
Thirty four published articles were included for data synthesis. A total of 1092 cases of odontogenic lesions were assessed for both CTNNB1 mutation and beta catenin expression. CTNNB1 mutation was observed in ameloblastoma, calcifying odontogenic cyst, calcifying cystic odontogenic tumour and all malignant odontogenic tumours. The beta catenin expression (nuclear and cytoplasmic) was maximum in odontogenic keratocyst and calcifying odontogenic cyst. The expression was variable in ameloblastomas, membranous in odontomas, calcifying cystic odontogenic tumour and nuclear in all malignant tumours.
DISCUSSION AND CONCLUSION
High recurrence of odontogenic keratocyst and aggressiveness of solid ameloblastoma and malignant odontogenic tumours could be associated with the nuclear translocation of beta catenin. Disparity between CTNNB1 mutation and beta catenin expression within odontogenic lesions suggests alternate routes of beta catenin activation. The review results support the unique localisation of beta catenin as a helpful diagnostic factor in the pathogenesis of odontogenic lesions.
Topics: Humans; Ameloblastoma; beta Catenin; Odontogenic Cyst, Calcifying; Odontogenic Cysts; Odontogenic Tumors
PubMed: 37840228
DOI: 10.1111/jop.13487 -
Head and Neck Pathology Sep 2023Recently, a new odontogenic tumor has been described, the so-called adenoid ameloblastoma (AdAM). The aim of this review was to determine the clinical and imaging...
BACKGROUND
Recently, a new odontogenic tumor has been described, the so-called adenoid ameloblastoma (AdAM). The aim of this review was to determine the clinical and imaging features of AdAM and to describe its main histopathological findings.
METHODS
The systematic review included published cases with a diagnosis of AdAM in the gnathic bones, which had sufficient clinical, imaging, and histopathological data to confirm its diagnosis. The following histopathological diagnostic criteria were adopted: presence of ameloblastoma-like components, duct-like structures, spiral cellular condensations, and a cribriform architecture.
RESULTS
Fifteen articles, corresponding to 30 cases of AdAM, were selected. Most cases affected men (63.3%), with a slight preference for the mandible (16:14) and the posterior region of gnathic bones was the most commonly affected site. The mean age at diagnosis was 40.8 years. Clinically, the lesions usually presented as a swelling (53.3%) and, radiographically, as a well-defined radiolucency (33.4%). Surgical resection (40%) was the most frequently adopted treatment and recurrence occurred in 30% of cases. Microscopic examination showed cribriform areas in most AdAM cases (93.3%); duct-like structures and spiral cellular condensations were seen in 100% of the cases.
CONCLUSION
The small number of reported cases, the existence of erroneous diagnoses, and the adoption of initial conservative management make it difficult to determine whether AdAM has a higher risk of recurrence or more aggressive biological behavior than conventional ameloblastomas.
Topics: Male; Humans; Adult; Ameloblastoma; Adenoids; Mandible; Odontogenic Tumors
PubMed: 37540485
DOI: 10.1007/s12105-023-01574-6 -
Journal of Oral Pathology & Medicine :... Sep 2023The objective of this systematic review with meta-analysis was to critically evaluate the available data on the association of the BRAF V600E mutation and recurrence... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
The objective of this systematic review with meta-analysis was to critically evaluate the available data on the association of the BRAF V600E mutation and recurrence rate of ameloblastomas.
MATERIALS AND METHODS
This systematic review was registered in Prospero (CRD42020183645) and performed based on the PRISMA statement. A comprehensive search in PubMed, Web of Science, Scopus and Cochrane Library databases was performed in order to answer the question "Does BRAF V600E mutation affect recurrence rate of ameloblastomas?" Methodological quality and risk of bias of the selected studies were assessed with JBI Critical Appraise Tool. Meta-analysis of quantitative data was conducted with RevMan 5.3 and Jamovi 2.3.
RESULTS
The initial search identified 302 articles, and 21 met the inclusion criteria. A total of 855 subjects with ameloblastoma were included in the analysis. The pooled measures for frequency of BRAF V600E mutation was 65.30% (95% CI: 0.56-0.75; p < .001; I = 90.85%; τ = 0.205; p < .001), and the pooled recurrence rate was 25.30% (95% CI: 0.19-0.31; p < .001; I = 79.44%; τ = 0.118; p < .001). No differences in recurrence rate were observed between the BRAF V600E and wild type BRAF ameloblastomas, with a pooled Odds Ratio of 0.93 (95% CI: 0.56-1.54; p = .78; I = 31%; p = .09).
CONCLUSIONS
BRAF V600E mutation is a frequent event in ameloblastomas, but does not increase nor reduce its recurrence rate, and thus have a limited value in predicting its prognosis.
Topics: Humans; Ameloblastoma; Proto-Oncogene Proteins B-raf; Mutation; Prognosis
PubMed: 37364158
DOI: 10.1111/jop.13458 -
Journal of Stomatology, Oral and... Oct 2022This meta-analysis provides reliable data on the prevalence of unicystic ameloblastomas (UA's) among solid/multicystic ameloblastomas (SMA's), as well the ratio of their... (Meta-Analysis)
Meta-Analysis
INTRODUCTION
This meta-analysis provides reliable data on the prevalence of unicystic ameloblastomas (UA's) among solid/multicystic ameloblastomas (SMA's), as well the ratio of their presence in the maxilla and mandible and in the tooth-bearing area versus the posterior regions of the mandible, including the third molar region and ascending ramus.
MATERIAL AND METHODS
A systematic review and meta-analysis was performed according to PRISMA guideline using the strategy ((unicystic ameloblastoma) OR (((ameloblastoma) OR (solid ameloblastoma)) OR (multicystic ameloblastoma))) NOT ((((systematic review) OR (literature review)) OR (case report)) OR (Immunohistochemical)).
DISCUSSION
The study included 3856 SMA's and 1537 UA's, which amounted to 28.5% UA's. Of the 380 cases of UA from twelve articles that mentioned the involved jaws, 355 were in the mandible and 25 in the maxilla. The preponderance for the mandible is much higher than reports from previous studies of smaller series. Only five articles mentioned the location within the mandible. The vast majority was in the posterior area.
CONCLUSION
The consequences for treatment were discussed, with an emphasis on the approach to unicystic lesions in the posterior part of the mandible for which a protocol is suggested.
Topics: Ameloblastoma; Head; Humans; Jaw; Mandible
PubMed: 35017129
DOI: 10.1016/j.jormas.2022.01.004 -
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 -
Frontiers in Oncology 2021This study aimed to systematically investigate and compare the post-treatment recurrence of intraosseous ameloblastoma in patients treated with conservative or...
OBJECTIVE
This study aimed to systematically investigate and compare the post-treatment recurrence of intraosseous ameloblastoma in patients treated with conservative or aggressive approaches.
METHODS
Systemic searches of PubMed, Medline, Cochrane Library, and Embase databases from inception to October 28, 2020, were conducted. Studies that aimed to evaluate the recurrence of intraosseous ameloblastoma by conservative and aggressive treatment approaches were included.
RESULTS
A total of 20 studies with 942 ameloblastoma cases were included. Fourteen studies included patients with ameloblastoma who received conservative treatment, and 16 studies reported the overall recurrence rate for patients undergoing aggressive treatment. The pooled results indicated that the recurrence rate for aggressive treatment [0.12, 95% confidence interval (CI) = 0.09-0.16] was significantly lower than that for conservative treatment, with a recurrence rate of 0.30 (95% CI = 0.23-0.39). Similar results were obtained when stratifying the participants by the histological classification. When trying stratification analysis following the original included studies, multicystic ameloblastoma presented a much higher recurrence rate than solid and unicystic ameloblastomas.
CONCLUSION
These findings supported the hypothesis that aggressive treatment might lead to a lower recurrence rate than conservative treatment. More studies and meta-analyses following the new histological classification of ameloblastomas are needed to validate and support the findings.
PubMed: 34094934
DOI: 10.3389/fonc.2021.647200 -
Oral Diseases May 2024Targeted therapy has the potential to be used in the neoadjuvant setting for odontogenic tumors, reducing the morbidities associated with major surgery. In this regard,... (Review)
Review
Targeted therapy has the potential to be used in the neoadjuvant setting for odontogenic tumors, reducing the morbidities associated with major surgery. In this regard, the aim of this study was to summarize the current evidence on the different forms of targeted therapy, effectiveness, and drawbacks of this course of treatment. Four databases were searched electronically without regard to publication date or language. Grey literature searches and manual searches were also undertaken. Publications with sufficient clinical data on targeted therapy for odontogenic tumors were required to meet the criteria for eligibility. The analysis of the data was descriptive. A total of 15 papers comprising 17 cases (15 ameloblastomas and 2 ameloblastic carcinomas) were included. Numerous mutations were found, with BRAF V600E being most common. Dabrafenib was the most utilized drug in targeted therapy. Except for one case, the treatment reduced the size of the lesion (16/17 cases), showing promise. Most of the adverse events recorded were mild, such as skin issues, voice changes, abnormal hair texture, dry eyes, and systemic symptoms (e.g., fatigue, joint pain, and nausea). It is possible to reach the conclusion that targeted therapy for ameloblastoma and ameloblastic carcinoma may be a useful treatment strategy, based on the findings of the included studies.
PubMed: 38693620
DOI: 10.1111/odi.14962 -
Journal of Conservative Dentistry : JCD 2022The objective of the study is to describe the clinical and radiographic features of nonmalignant nonendodontic periapical lesions (NMNPLs) mimicking lesions of... (Review)
Review
The objective of the study is to describe the clinical and radiographic features of nonmalignant nonendodontic periapical lesions (NMNPLs) mimicking lesions of endodontic cause. Five electronic databases, PubMed, Web of Science, Scopus, Embase, and ProQuest, were searched (till July 2021) for case reports, case series, and cross-sectional studies, in English language, reporting NMNPLs, which were clinically and/or radiographically simulating periapical pathosis of endodontic origin. Data extraction was done followed by quality assessment of the included articles using the Joanna Briggs Institute tool for case reports and case series. Seventy-three articles comprising 176 cases were included. Sixty-one articles were case reports, nine articles were case series, and three articles were retrospective studies. Male:female ratio was 1.5:1, with a higher prevalence of lesions occurring in the fourth and second decades of life. The majority of the lesions were located in the anterior maxilla. Radiographically, most of the lesions were well defined, radiolucent, and unilocular. Histologically, 29 different types of NMNPLs were reported, with the most common ones being odontogenic keratocyst (25.56%), dentigerous cyst (17.61%), ameloblastoma (11.36%), nasopalatine duct cyst (10.79%), and adenomatoid odontogenic tumor (5.68%). As all the included studies were observational, the quality of available evidence is considered low. Various features such as loss of tooth vitality, history of trauma, and presence of periapical radiolucency may lead to misdiagnosis of NMNPLs and must be considered during diagnosis of the lesion. Additional imaging modalities and histopathology can aid in right diagnosis.
PubMed: 35836562
DOI: 10.4103/jcd.jcd_13_22