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Cureus Oct 2023This article was previously posted to the Research Square preprint server on 16 August 2023. Maxillary sinus ameloblastoma is an uncommon, locally aggressive odontogenic...
This article was previously posted to the Research Square preprint server on 16 August 2023. Maxillary sinus ameloblastoma is an uncommon, locally aggressive odontogenic tumor. In this case report, we present a comprehensive long-term follow-up of maxillary sinus ameloblastoma occurring in a patient with Turner syndrome, managed through partial right maxillectomy followed by adjuvant operative bed radiotherapy.
PubMed: 38022157
DOI: 10.7759/cureus.47808 -
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 -
Cureus Oct 2023Keratocystic odontogenic tumor (KCOT) is a benign intraosseous cyst of the jaw. The diagnosis may be confirmed by the clinical finding, or histopathological report....
Keratocystic odontogenic tumor (KCOT) is a benign intraosseous cyst of the jaw. The diagnosis may be confirmed by the clinical finding, or histopathological report. Finally, treatment consists of surgical excision, and follow-up is characterized by a minor recurrence. The authors report a case of KCOT of the right mandibular region and review the various diagnoses, therapeutics, and follow-up aspects of this type of tumor. The following case report describes a case of a 55-year-old male patient with odontogenic keratocyst. It can show variable clinical-radiographic features and hence should be in differential diagnosis of intra-osseous oral lesions in old age. In elderly patients, because of physical disability, selection of treatment procedures and long-term follow-up after surgery is vital.
PubMed: 38021646
DOI: 10.7759/cureus.46963 -
Medicina (Kaunas, Lithuania) Nov 2023Ameloblastoma is the most common benign odontogenic tumor with local invasion and high recurrence, which generally occurs in the jaw bones. Hypercalcemia is a common... (Review)
Review
Ameloblastoma is the most common benign odontogenic tumor with local invasion and high recurrence, which generally occurs in the jaw bones. Hypercalcemia is a common paraneoplastic syndrome that is commonly observed in patients with malignancies but rarely encountered in patients with benign tumors. Thus far, not many cases of ameloblastoma with hypercalcemia have been reported, and the pathogenic mechanism has not been studied in depth. This paper presents a case report of a 26-year-old male diagnosed with giant ameloblastoma of the mandible, accompanied by rare hypercalcemia. Additionally, a review of the relevant literature is conducted. This patient initially underwent marsupialization, yet this treatment was not effective, which indicated that the selection of the appropriate operation is of prime importance for improving the prognosis of patients with ameloblastoma. The tumor not only failed to shrink but gradually increased in size, accompanied by multiple complications including hypercalcemia, renal dysfunction, anemia, and cachexia. Due to the contradiction between the necessity of tumor resection and the patient's poor systemic condition, we implemented a multi-disciplinary team (MDT) meeting to better evaluate this patient's condition and design an individualized treatment strategy. The patient subsequently received a variety of interventions to improve the general conditions until he could tolerate surgery, and finally underwent the successful resection of giant ameloblastoma and reconstruction with vascularized fibular flap. No tumor recurrence or distance metastasis was observed during 5 years of follow-up. Additionally, the absence of hypercalcemia recurrence was also noted.
Topics: Male; Humans; Adult; Ameloblastoma; Hypercalcemia; Mandibular Neoplasms; Neoplasm Recurrence, Local; Mandible
PubMed: 38004005
DOI: 10.3390/medicina59111956 -
BMC Oral Health Nov 2023Patients with mandibular conventional ameloblastoma undergoing radical surgical treatment experience greater trauma and often find it challenging to accept, whereas...
BACKGROUND
Patients with mandibular conventional ameloblastoma undergoing radical surgical treatment experience greater trauma and often find it challenging to accept, whereas conservative therapy is associated with a higher recurrence rate. In this study, we have improved traditional conservative treatment for mandibular conventional ameloblastoma by curettage combined with bone cavity opening (Cur/BCO). This retrospective study aimed to evaluate the effectiveness of the Cur/BCO treatment by comparing its recurrence rate and bone mineral density (BMD) growth rate with the traditional conservative treatment approach.
METHODS
A total of 40 patients, meeting the study's inclusion and exclusion criteria from 2012 to 2020, were screened, with 20 in the modified group and 20 in the traditional group. ImageJ (RRID: SCR_003070) software was employed for measuring image indices. All data were analyzed using T-test, Chi-square test and Fisher exact test in SPSS 26.0 (p = 0.05).
RESULTS
The incidence of recurrence was significantly lower in the modified group, at only 5%, compared to 35% in the traditional group (p < 0.05). Regarding bone mineral density (BMD) growth rate, the average value in the modified group was 0.0862 ± 0.2302 (/month), significantly higher than the average value of 0.0608 ± 0.2474 (/month) in the traditional group (p < 0.05).
CONCLUSIONS
In this study, it was found that the recurrence rate of the modified conservative treatment (Cur/BCO) was lower than that of the traditional conservative treatment for managing mandibular conventional ameloblastoma. Furthermore, the BMD growth rate was quicker in the modified group. Thus, Cur/BCO could be considered as a viable option for the conservative treatment of mandibular conventional ameloblastoma.
Topics: Humans; Ameloblastoma; Retrospective Studies; Mandibular Neoplasms; Neoplasm Recurrence, Local; Mandible; Curettage
PubMed: 37986074
DOI: 10.1186/s12903-023-03660-1 -
Journal of Advanced Veterinary and... Sep 2023The escalating prevalence of canine oral tumors has emerged as a considerable health concern. This study examined the prevalence, types, and distributions of lesions...
OBJECTIVE
The escalating prevalence of canine oral tumors has emerged as a considerable health concern. This study examined the prevalence, types, and distributions of lesions linked to canine oral tumors.
MATERIAL AND METHODS
The medical records of 526 dogs diagnosed with oral tumors were analyzed to determine the prevalence, types, and distributions. Tumor stages were classified into four categories using the tumor node metastasis system.
RESULTS
Among the 526 dogs, there were 118 cases of benign tumors and 408 cases of malignant tumors. Acanthomatous ameloblastoma was the most common benign tumor (43.22%), while melanoma was the most common malignant tumor (51.23%). The gingiva was the most common site for both benign and malignant lesions, accounting for 89.83% and 63.73% of cases, respectively. Melanoma, squamous cell carcinoma, and fibrosarcoma were primarily located in the gingiva, whereas osteosarcoma was commonly found in the mandible. Most tumors were classified as stage III (ranging from 46.84% to 74.58%). Of the reported cases, 56.08% were males and 43.92% were females, and the most common breed was mixed at 30.41%, followed by Poodle at 14.25% and Shih Tzu at 11.40%. Moreover, patients with malignant oral tumors (11.6 ± 3.1 years) were significantly older than those with benign tumors (8.9 ± 3.4 years, < 0.0001).
CONCLUSION
Gingiva was the primary site for oral tumors, and mainly classified as stage III. These findings emphasize the increasing occurrence of oral tumors in senior and geriatric dogs and provide insights into the prevalent types and distribution.
PubMed: 37969809
DOI: 10.5455/javar.2023.j709 -
Case Reports in Oncology 2023Ameloblastic fibrosarcoma (AFS) is considered a malignant progression resulting from dysplastic changes in an ameloblastic fibroma (AF). Both tumors are extremely rare,...
Ameloblastic fibrosarcoma (AFS) is considered a malignant progression resulting from dysplastic changes in an ameloblastic fibroma (AF). Both tumors are extremely rare, with only a few cases reported in the scientific literature. Notably, BRAF mutations have been identified in ameloblastomas, suggesting a connection between ameloblastic morphology and BRAF mutations, as AF is believed to be the precursor neoplasm leading to AFS. In this study, we present a case of AFS in a 25-year-old male. The tumor tissue underwent molecular analysis, specifically next-generation sequencing (NGS) using the Oncomine Comprehensive Assay v3 System. The analysis revealed pathogenic mutations in TP53 and RB genes, as well as copy number gains in NTRK1, MDM4, and BRAF. Additionally, we provide a summary of the literature's findings from the analysis of 107 previously reported AFS cases. Our findings suggest the existence of a molecularly distinct subtype, emphasizing the importance of comprehensive molecular testing for these patients.
PubMed: 37942402
DOI: 10.1159/000532014 -
Cancer Diagnosis & Prognosis 2023Tumors and cysts with odontogenic origin represent a family of lesions with specific histo-genetic and clinical characteristics. Among them, ameloblastomas are common... (Review)
Review
Tumors and cysts with odontogenic origin represent a family of lesions with specific histo-genetic and clinical characteristics. Among them, ameloblastomas are common benign neoplasms, predominantly detected in the anatomic areas of the jaws and also in the mandible and maxilla. Although they are characterized by a slow and stable growing pattern, a subset of them shows a tendency for local tissue invasiveness and partially increased recurrence rates after surgical excision. Furthermore, heat shock proteins (HSPs) are potentially implicated in ameloblastoma onset and progression. HSPs regulate the folding and refolding of proteins and are induced in response to oxidative stress. They are crucial members of the chaperone intracellular system and are categorized based on their molecular weight (i.e., HSP27, HSP60, HSP70, HSP90). In the current review, we describe HSPs origin and function, focusing on their deregulation mechanisms and impact predominantly on ameloblastomas and also on inflammatory and developmental odontogenic cystic lesions.
PubMed: 37927807
DOI: 10.21873/cdp.10265 -
Frontiers in Veterinary Science 2023Differentiating canine acanthomatous ameloblastoma (CAA) from oral squamous cell carcinoma (OSCC) based on routine histopathology can be challenging. We have previously...
Differentiating canine acanthomatous ameloblastoma (CAA) from oral squamous cell carcinoma (OSCC) based on routine histopathology can be challenging. We have previously shown that more than 95% of CAAs harbor an p.Q61R somatic mutation, while OSCCs carry either wild-type alleles or other MAPK pathway activating mutations (e.g., p.Q61L, p.V595E). Given that p.Q61R mutations are highly prevalent in CAA, we hypothesized that a RAS Q61R-specific rabbit monoclonal antibody may be a useful tool for confirmation of CAA by immunohistochemical (IHC) staining. In the present study, we assessed IHC staining of archived formalin-fixed and paraffin-embedded biopsy samples with a diagnosis of CAA ( = 23), using a RAS Q61R-specific rabbit monoclonal antibody (SP174) and an automated IHC stainer. Negative control samples consisted of p.Q61R mutation-negative OSCC tumors with either a known p.Q61L mutation ( = 1), p.V595E mutation ( = 4), or wild-type corresponding alleles ( = 3). We found that all 23 CAAs showed diffuse and strong membranous RAS Q61R immunoreactivity (100% sensitivity), while none of the 8 OSCCs showed immunoreactivity (100% specificity). The data supports the use of RAS Q61R-specific rabbit monoclonal antibody for diagnostic IHC confirmation of CAA and ruling out OSCC in dogs.
PubMed: 37901104
DOI: 10.3389/fvets.2023.1281022 -
Journal of Oral and Maxillofacial... 2023The purpose of this experimental study was to evaluate and compare the degree of expression of Wilm's Tumor Gene-1 (WT-1), Syndecan (CD 138) and Snail in Ameloblastoma...
BACKGROUND
The purpose of this experimental study was to evaluate and compare the degree of expression of Wilm's Tumor Gene-1 (WT-1), Syndecan (CD 138) and Snail in Ameloblastoma and odontogenic keratocyst (OKC) and to analyse their potential role in pathogenesis.
METHODS AND MATERIAL
Immunohistochemical analysis was performed to evaluate WT-1, Syndecan and Snail expression in Ameloblastoma ( = 20) and OKC ( = 20). Topographical immunoexpression pattern of Ameloblast-like cells, Stellate Reticulum-like cells in Ameloblastoma and basal layer as well as suprabasal layer of cells of OKC were also compared. The results obtained were subjected to ANOVA test and Tukey HSD test through SPSS software 20.0 for Microsoft Windows.
RESULTS
WT-1 and Snail overexpression was seen in both Ameloblastoma and OKCs. Syndecan, responsible for maintaining normal cellular morphology, cell-cell adhesion and differentiation was significantly downregulated in both the lesions. The Ameloblasts-like cells and the basal cells showed significantly higher immunopositivity for WT-1 and Syndecan as compared to that of basal cells. An inverse relation was noted for Snail protein. The ANOVA test predicted a statistically significant difference of expression across the lesions with a value <0.0001 for Syndecan and Snail.
CONCLUSIONS
The under-expression of epithelial membrane protein Syndecan-1 and upregulation of EMT transcription factor Snail can promote local invasion and is indicative of poor prognosis of these lesions. The overexpression of WT-1 results in tumorigenesis, proliferation and localized aggressiveness of Ameloblastoma and intrabony growth of OKC. Further investigation on the biologic behaviour of OKC is still recommended to arrive at more specific conclusions regarding its nature.
PubMed: 37854929
DOI: 10.4103/jomfp.jomfp_301_22