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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 Oral Pathology & Medicine :... Mar 2024Ameloblastic carcinoma (AC) is the most common odontogenic malignancy, constituting approximately 30% of cases in this category. Literature is sparse on malignant... (Review)
Review
BACKGROUND
Ameloblastic carcinoma (AC) is the most common odontogenic malignancy, constituting approximately 30% of cases in this category. Literature is sparse on malignant odontogenic neoplasms, with a large proportion of current knowledge derived from case reports or small case series.
METHODS
A systematic review of case series/case reports of AC was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) Statement guidelines. Demographic and clinical information, including duration of the lesion, location, clinical presentation and radiologic features, were analysed. Additionally, the origin of the lesion (primary/secondary), Ki-67 proliferation index, treatment performed, metastasis, tumour recurrence and prognosis were collected for analysis.
RESULTS
A total of 126 studies, including 285 individual cases of AC, were included in this review. Patients presented with a near-equal distribution of painless and painful swellings. ACs presented at a median age of 45 years, with a male-to-female ratio of 1:2. The mandible was most frequently involved, with rare cases extending to involve more than one region, including crossing the midline. Although most lesions presented with poorly-demarcated borders (52.6%), unilocular lesions with well-demarcated borders (47.4%) comprised a substantial number in the sample. The proliferation index was only reported in 27 cases, with a mean score of 42% and a wide range. The probability of tumour recurrence increased, and the survival probability decreased with prolonged follow-up duration.
CONCLUSION
This study provides more comprehensive, up-to-date descriptive data on these rare odontogenic malignancies, aiding clinicians and Pathologists with the diagnosis and surgeons in their management of cases.
Topics: Humans; Male; Female; Middle Aged; Neoplasm Recurrence, Local; Odontogenic Tumors; Mandible; Prognosis; Carcinoma
PubMed: 38368851
DOI: 10.1111/jop.13517 -
BMC Oral Health Dec 2023Ameloblastoma (AM), the benign counterpart of ameloblastic carcinoma, is a benign odontogenic tumor of epithelial origin, naturally aggressive, with unlimited growth...
BACKGROUND
Ameloblastoma (AM), the benign counterpart of ameloblastic carcinoma, is a benign odontogenic tumor of epithelial origin, naturally aggressive, with unlimited growth potential and a high tendency to relapse if not adequately removed. Patients with AM treated surgically can benefit from dental implant therapy, promoting oral rehabilitation and improving their quality of life. The present study aimed to determine the survival rate of dental implants placed after surgical treatment of patients affected by AM. In addition, there were two secondary objectives: 1) To evaluate which dental implant loading protocols are most frequently used and 2) To determine the type of prosthetic restoration most commonly used in these patients.
METHODS
The Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA) guidelines were followed during the study. Searches were performed in three databases (PubMed/MEDLINE, Scopus, and Google Scholar) until November 2023. Additionally, the electronic search was enriched by an iterative hand search of journals related to oral pathology and medicine, maxillofacial surgery, and oral prosthodontics and implantology. Only reports and case series in English from January 2003 to date were included. The Joanna Briggs Institute tool (JBI-Case Reports/Case Series) was used for the study quality assessment.
RESULTS
The total number of patients and implants studied were 64 and 271, respectively, all with surgically treated AM. The patient's ages ranged from 8 to 79 years, with a mean (SD) age of 37.3 ± 16.4. Fifty-three percent were male and 47% were female. The range of follow-up duration was 1 to 22 years. An implant survival/success rate of 98.1% was reported. In addition, most of them were conventionally loaded (38.3%). Hybrid implant-supported fixed dentures were the most commonly used by prosthodontists (53%).
CONCLUSIONS
Oral rehabilitation with dental implants inserted in free flaps for orofacial reconstruction in surgically treated patients with AM can be considered a safe and successful treatment modality.
Topics: Adolescent; Adult; Aged; Child; Female; Humans; Male; Middle Aged; Young Adult; Ameloblastoma; Dental Implantation, Endosseous; Dental Implants; Dental Prosthesis, Implant-Supported; Dental Restoration Failure; Neoplasm Recurrence, Local; Odontogenic Tumors; Quality of Life; Treatment Outcome
PubMed: 38110933
DOI: 10.1186/s12903-023-03765-7 -
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 -
Asian Pacific Journal of Cancer... Nov 2022Ameloblastoma is regarded as the second most prevalent odontogenic tumor in the light of its prevalence, clinical characteristics, greater incidence of tumor recurrence,... (Meta-Analysis)
Meta-Analysis
INTRODUCTION
Ameloblastoma is regarded as the second most prevalent odontogenic tumor in the light of its prevalence, clinical characteristics, greater incidence of tumor recurrence, and therapeutic challenges. The aim of this systematic review was to establish the prevalence of ameloblastoma in the Indian subcontinent and to establish a national epidemiologic profile for these lesions.
MATERIAL AND METHODS
A systematic review was undertaken based on the PRISMA guidelines in search of epidemiologic studies concerning odontogenic tumors and ameloblastoma that are listed by PubMed, EBSCO, and Google Scholar embracing the period from January 2010 to December 2021, to evaluate the prevalence rate in India. A total of 277 publications were retrieved, of which 27 articles were selected, based on the World Health Organization classification of odontogenic tumors.
RESULTS
The affected individuals were on average in the third decade of life, with a higher male predominance. The majority of the tumors were multilocular radiolucencies in the posterior mandible, with follicular and plexiform histopathological features. The most common type of malignant lesion is ameloblastic carcinoma. Over 60% of follicular ameloblastoma recurred more frequently than the other types of ameloblastoma.The random effect model shows overall point estimate of 4.83 with 95% confidence interval (4.44 -5.26).
CONCLUSION
The systematic study indicates a slight male predisposition to ameloblastoma, with a peak incidence in the third decade of life and the mandible as the preferred anatomical site. The solid/multicystic ameloblastoma is the most prevalent histopathologic pattern. More epidemiological research on the prevalence rate of ameloblastoma is required, particularly in India, in an effort to accurately determine the national epidemiological profile of ameloblastoma.
Topics: Male; Humans; Female; Ameloblastoma; Prevalence; India; Odontogenic Tumors; Genotype
PubMed: 36444570
DOI: 10.31557/APJCP.2022.23.11.3601 -
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 -
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 -
Journal of Stomatology, Oral and... Apr 2021Ameloblastoma is a benign odontogenic tumor which undergoes malignant transformation to ameloblastic carcinoma. However, rarely it metastasizes without undergoing...
Ameloblastoma is a benign odontogenic tumor which undergoes malignant transformation to ameloblastic carcinoma. However, rarely it metastasizes without undergoing cytological malignant changes, an entity referred to as Metastasizing Ameloblastoma (MA). Through this study, we aimed to review cases of MA reported since 2000 to explore the impact of clinico-demographic variables on its prognosis. Based on PRISMA guidelines, a review of relevant literature from PubMed/Medline, Science Direct and Cochrane database was performed from January 2000 to March 2019. A total of 65 cases were considered for further evaluation as per predefined inclusion and exclusion criteria. Results showed that lungs followed by lymph nodes were the most common sites for benign metastatic deposits. Multiple recurrences and inadequate surgical removal increase the probability of distant metastatic spread. Despite having benign cytological features, tumor recurrence and metastasis were associated with an unfavorable clinical outcome in MA.
Topics: Ameloblastoma; Cell Transformation, Neoplastic; Humans; Neoplasm Recurrence, Local; Odontogenic Tumors; Prognosis
PubMed: 32659412
DOI: 10.1016/j.jormas.2020.07.001 -
World Journal of Clinical Oncology Jan 2020Ameloblastomas are common benign epithelial odontogenic neoplasms that present an aggressive and unpredictable behavior that may modify treatment strategies. Different...
BACKGROUND
Ameloblastomas are common benign epithelial odontogenic neoplasms that present an aggressive and unpredictable behavior that may modify treatment strategies. Different signaling pathways that participate in the progression of these tumors have been identified. B-raf proto-oncogene serine/threonine kinase (BRAF) is a protein involved in the behavior of ameloblastomas, and it is related to many cell mechanisms. BRAF gene mutations have been identified in ameloblastomas, of which the BRAF V600E (valine substituted by glutamic acid at amino acid 600) mutation has been the most common and can be present concomitantly with other mutations that may be involved in its behavior. Targeted therapies have been used as an alternative in the case of resistance or contraindications to conventional treatments.
AIM
To document the presence of BRAF V600E and additional mutations, their behavior, and targeted therapies in these tumors.
METHODS
An electronic literature search was conducted according to PRISMA guidelines in PubMed/MEDLINE, Cochrane, EMBASE, and SpringerLink using the terms "ameloblastomas", "BRAF V600E", "additional mutations", and "targeted therapies". Ameloblastomas were classified according to WHO guidelines. Inclusion criteria were articles in English, published not more than 10 years ago, and studies with laboratory works related to BRAF V600E. Articles were evaluated by two independent reviewers and retrieved for full-text evaluation. The EBLIP Critical Appraisal Checklist was used to evaluate the quality of the eligible studies. Descriptive statistical analysis was performed.
RESULTS
Two independent reviewers, with a substantial concordance indicated by a kappa coefficient of = 0.76, evaluated a total of 19 articles that were included in this study. The analysis registered 521 conventional ameloblastomas (AM), 81 unicystic ameloblastomas (UA), 13 ameloblastic carcinomas (AC), three metastatic ameloblastomas (MA), and six peripheral ameloblastomas (PA), of which the histopathological type, anatomic location, laboratory tests, expression of BRAF mutation, and additional mutations were registered. The BRAF V600E mutation was found in 297 AM (57%), 63 UA (77.7%), 3 AC (23%), 1 MA (50%), and 5 PA (83.3%). Follicular type predominated with a total of 116 cases (40%), followed by plexiform type with 63 cases (22.1%). Furthermore, both types presented additional mutations, in which alterations in JAK3 P132T, SMARCB1, PIK3CA, CTNNB1, SMO, and BRAF G606E genes were found. Four case reports were found with targeted therapy to BRAF V600E.
CONCLUSION
The identification of BRAF V600E and additional mutations as an aid in targeted therapies has been a breakthrough in alternative treatments of ameloblastomas where surgical treatments are contraindicated.
PubMed: 31976308
DOI: 10.5306/wjco.v11.i1.31 -
Head & Neck Dec 2019Exploring the clinicopathological features of ameloblastic carcinoma (AC) and reviewing the literature to improve the diagnosis and treatment of the disease.
OBJECTIVES
Exploring the clinicopathological features of ameloblastic carcinoma (AC) and reviewing the literature to improve the diagnosis and treatment of the disease.
MATERIALS AND METHODS
Clinical data and pathological features of 18 cases of AC were retrospectively analyzed. A systematic review was carried out by searching PubMed and Medline databases using the MeSH terms "ameloblastic" and "carcinoma."
RESULTS
In the systematic analysis, 125 cases of AC from 81 eligible original studies and 18 cases of AC from this research were included. The male-to-female ratio was 2.58:1, and the mandible-to-maxilla ratio was 1.80:1. Mean age of patients was 45.3 years. Thirty-seven cases of recurrence and 27 cases of metastasis were recorded.
CONCLUSION
AC is a rare neoplasm of the odontogenic epithelium. A systematic review indicates that diagnoses at the early phase and a close periodic assessment for recurrence and metastasis are necessary.
Topics: Adult; Aged; Ameloblastoma; Carcinoma; Female; Humans; Male; Mandibular Neoplasms; Maxillary Neoplasms; Middle Aged; Neoplasm Metastasis; Neoplasm Recurrence, Local; Odontogenic Tumors; Retrospective Studies; Survival Rate
PubMed: 31444935
DOI: 10.1002/hed.25926