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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 -
Medicina (Kaunas, Lithuania) Jun 2020Plexiform ameloblastoma is a locally aggressive odontogenic tumor, rare in the anterior mandible. The treatment of choice is resection with 1-3 cm free margins. In most...
BACKGROUND
Plexiform ameloblastoma is a locally aggressive odontogenic tumor, rare in the anterior mandible. The treatment of choice is resection with 1-3 cm free margins. In most of reported cases, the affected mandible is reconstructed by autogenic bone graft or osseocutaneous microvascular free flap in order to return function and esthetics.
CASE DESCRIPTION
A 2 cm diameter exophytic ameloblastoma, located in the anterior mandible of a 50-year-old male was resected and reconstructed in a unique manner-allogenic bone block, recombinant human bone morphogenetic protein (rhBMP) and xenograft particles via transcutaneous submental approach. After bone maturation, dental implants were placed and restored by fixed prosthetics.
PRACTICAL IMPLICATIONS
Mandible reconstruction modalities have a crucial influence on patient quality of life, function and esthetics. Allogenic bone block combined with rhBMP and xenograft particles can replace the traditional autogenous bone in certain circumstances. A submental transcutaneous "tent pole" approach can improve the success rate of the reconstruction procedure.
Topics: Bone Transplantation; Humans; Male; Mandible; Mandibular Osteotomy; Middle Aged; Neurofibroma, Plexiform; Plastic Surgery Procedures
PubMed: 32630080
DOI: 10.3390/medicina56070326 -
Brazilian Oral Research 2022The osteolytic activity of odontogenic cysts and tumors is directly associated with their growth and aggressiveness. The influence of proteins expressed by epithelial...
The osteolytic activity of odontogenic cysts and tumors is directly associated with their growth and aggressiveness. The influence of proteins expressed by epithelial and mesenchymal cells on this biological event differs between indolent cystic lesions, aggressive cystic lesions, and odontogenic tumors. The objective of this study was to compare the immunohistochemical expression of factors that stimulate (receptor activator of nuclear factor kappa-Β ligand - RANKL, cathepsin K - CatK and matrix metallopeptidase 8 - MMP-8) and inhibit (osteoprotegerin - OPG) osteoclastogenesis between dentigerous cyst (DC), glandular odontogenic cyst (GOC), odontogenic keratocyst (OKC), and ameloblastoma (AB). Paraffin-embedded sections of nine DCs, nine GOCs, 20 OKCs, 21 ABs, and four dental follicles (DFs) were subjected to immunohistochemistry. Immunoreactivity was analyzed semiquantitatively and quantitatively in epithelium and connective tissue, respectively. The proteins were immunoexpressed in epithelial and mesenchymal cells of all lesions studied. The expression of RANKL and CatK was higher in OKC, AB, and GOC (p<0.005). Higher expression of OPG was found in DF and DC compared to the other markers (p<0.005). MMP-8 expression was high in GOC and OKC. This study demonstrated the differential expression of factors that inhibit and stimulate bone resorption during the development of DC, GOC, OKC, and AB. Higher expression of RANKL and CatK was observed in more aggressive lesions. OPG appears to be one of the molecules responsible for the slower growth of DC.
Topics: Humans; Dentigerous Cyst; Matrix Metalloproteinase 8; Odontogenic Cysts; Ameloblastoma; Odontogenic Tumors
PubMed: 36507759
DOI: 10.1590/1807-3107bor-2022.vol36.0072 -
Archives of Oral Biology May 2023To perform an integrated analysis in identifying novel hub genes that could facilitate the diagnosis and targeted therapy of ameloblastoma.
OBJECTIVE
To perform an integrated analysis in identifying novel hub genes that could facilitate the diagnosis and targeted therapy of ameloblastoma.
DESIGN
The expression profiling dataset, GSE38494, was obtained from the Gene Expression Omnibus database. Differentially expressed genes were identified through GEO2R online tool and characterised via Gene Ontology (GO) analysis and Kyoto Encyclopedia of Genes and Genomes (KEGG) pathways. The protein-protein interaction network and hub genes were screened using the STRING database and Cytoscape software. Subsequently, an upregulated gene was selected for further validation using the GSE132472 dataset. Further, immunohistochemistry was performed to assess the expression of the selected gene in ameloblastomas, odontogenic keratocysts, dentigerous cysts, and gingival tissues. The diagnostic and therapeutic utility of the selected hub genes were further verified by receiver operating characteristic analysis and the DGIdb database.
RESULTS
We identified six hub genes in ameloblastoma, among which the upregulated gene PKD2 and its related gene PKD1 were further validated. GO functional annotation revealed that PKD2 is involved in cell-cell junction, extracellular exosome, cytoplasm, endoplasmic reticulum, and calcium ion transport. The immunohistochemical analysis showed that the expression of polycystin-1 and polycystin-2, encoded by the PKD1 and PKD2 genes, respectively, was upregulated in ameloblastoma. PKD1 and PKD2 had a high diagnostic utility for ameloblastoma, and allopurinol interacted with the PKD2 gene.
CONCLUSION
Our research indicates that polycystins are highly expressed in ameloblastoma and might be involved in the oncogenesis of ameloblastoma, thus offering a new perspective on the molecular mechanisms and targeted therapies on ameloblastoma.
Topics: Humans; Ameloblastoma; Gene Expression Profiling; TRPP Cation Channels; Biomarkers, Tumor; Immunohistochemistry; Computational Biology; Gene Expression Regulation, Neoplastic
PubMed: 36857877
DOI: 10.1016/j.archoralbio.2023.105662 -
BMC Oral Health Mar 2022To explore the masticatory performance in patients undergoing an osteo(cutaneous) free fibula (OFF) flap for mandible reconstruction by a prospective design.
BACKGROUND
To explore the masticatory performance in patients undergoing an osteo(cutaneous) free fibula (OFF) flap for mandible reconstruction by a prospective design.
METHODS
A total of 56 patients who had undergone OFF flap reconstructions for mandibular reconstruction secondary to malignant (squamous cell carcinoma) or benign (ameloblastoma) tumor resection were prospectively enrolled. They were asked to complete the masticatory performance test by the weigh method and the chew domain of the University of Washington quality of life questionnaire (version 4) preoperatively and at 3, 6, and 12 months postoperatively. The pair nonparametric test was used to analyze the dynamic change of masticatory performance and subjective chew function.
RESULTS
Fifty-one patients were included for analysis finally. The mean masticatory performance for patients with malignant tumors were 53.4% ± 10.3%, 36.4% ± 10.3%, 42.6% ± 9.6%, 52.8% ± 10.9%, and 53.1% ± 11.8% preoperatively, at 2 weeks, 3 months, 6 months, and 12 months postoperatively, respectively. Compared with the preoperative level, the masticatory performance had a significant reduction immediately after surgery (p < 0.001), followed by a return to the baseline level within three months. A similar trend was noted for those with benign tumors. The mean score of chew domain for patients with malignant tumors were 100 ± 0, 54.3 ± 32.9, 81.4 ± 24.5, and 92.9 ± 17.8 preoperatively, at 3 months, 6 months, and 12 months postoperatively, respectively. Compared with the preoperative level, the subjective chew function was greatly affected within the first three months (p < 0.001), and it gradually recovered to the baseline level in the following nine months. A similar trend was noted in patients with benign tumors.
CONCLUSIONS
The masticatory performance and subjective chew function was significantly affected after OFF flap reconstructions in the short term, but both recovered to the preoperative levels within 9-12 months.
Topics: Ameloblastoma; Fibula; Free Tissue Flaps; Humans; Mandible; Mandibular Reconstruction; Prospective Studies; Quality of Life
PubMed: 35300661
DOI: 10.1186/s12903-022-02114-4 -
International Journal of Surgery Case... Dec 2022Ameloblastomas are slow growing and locally aggressive odontogenic tumors with a high propensity for recurrence. It frequently arises in the mandible and has been...
INTRODUCTION
Ameloblastomas are slow growing and locally aggressive odontogenic tumors with a high propensity for recurrence. It frequently arises in the mandible and has been reported to metastasize commonly in the lungs. An updated World Health Organization classification re-categorized metastasizing ameloblastomas under benign tumors. Other rare metastatic sites include the skull, maxilla, kidney, and liver.
CASE PRESENTATION
We present a 53-year-old female with a gradually enlarging right breast mass for 2 years. She previously underwent right hemimandibulectomy with clavicular bone grafting 15 years ago for a primary ameloblastoma. Preoperative imaging showed a resectable, heterogenous right breast mass with a biopsy revealing spindle cell neoplasm. She subsequently underwent radical mastectomy with a latissimus dorsi myocutaneous flap as a reconstructive procedure. Histopathologic findings were consistent with a metastasizing ameloblastoma. The patient remains disease-free as of most recent follow-up.
DISCUSSION
There are several proposed mechanisms for metastasizing ameloblastomas. Based on the history and location of the tumor, we surmised that tumor seeding from the first surgery done 15 years ago may explain this rare occurrence. Preoperative imaging and biopsy determine resectability and surgical approach. Radical surgery is frequently performed which largely depends on the site of the tumor. Complete primary resection with adequate margins remains to be the treatment of choice to prevent recurrence or metastasis. The role of adjuvant radiotherapy or chemotherapy are still to be established.
CONCLUSION
This case highlights the value of history-taking and having a high-index of suspicion for metastasis several years after primary resection of ameloblastomas.
PubMed: 36436420
DOI: 10.1016/j.ijscr.2022.107800 -
Acta Bio-medica : Atenei Parmensis May 2023The purpose of this study was to determine the clinical and histological features and treatment of peripheral ameloblastoma. Peripheral Ameloblastoma is a rare benign...
BACKGROUND AND AIM
The purpose of this study was to determine the clinical and histological features and treatment of peripheral ameloblastoma. Peripheral Ameloblastoma is a rare benign odontogenic tumor that concerns soft tissue and have a typical extraosseous localization.
METHODS
Aim of this work is to show its clinical and histological characters, in order to define some useful information for differential diagnosis with other oral neoformations, comparing literature with our data, collected in ten years of clinical activity of Oral and Maxillofacial Surgery Unit of Policlinico Tor Vergata in Rome.
RESULTS
Prognosis of PA is certainly favourable, with a restitutio ad integrum close to 100%. In the period between October 2011 and November 2021, we reported 8 diagnoses of P.A. Medium age of the group with diagnosis of PA was 71,4 y with a SD: 3,65. P.A.'s incidence in our sample of patients was 0,26%.
CONCLUSIONS
PA is a benign odontogenic tumor that requires a careful diagnosis, a complete surgical eradication and adequate follow up, because malignant evolution is rare but possible.
Topics: Humans; Ameloblastoma; Odontogenic Tumors; Prognosis; Diagnosis, Differential; Incidence
PubMed: 37213074
DOI: 10.23750/abm.v94iS1.13527 -
Dento Maxillo Facial Radiology Sep 2019To characterize the radiographic features of maxillary ameloblastoma (AM), odontogenic keratocyst (OKC) and dentigerous cyst (DC) comparatively by using spiral CT and...
OBJECTIVES
To characterize the radiographic features of maxillary ameloblastoma (AM), odontogenic keratocyst (OKC) and dentigerous cyst (DC) comparatively by using spiral CT and cone beam CT (CBCT).
METHODS
Clinical records, histopathological reports, and nonenhanced spiral CT or CBCT images of 191 consecutive patients with primary maxillary AMs, OKCs, or DCs were retrospectively acquired, and radiographic features were analyzed.
RESULTS
The study included 118 males and 73 females (age: 5-84 years). 72.0% of AMs and 84.3% of OKCs originated from the posterior maxilla, while 69.6% of DCs occurred in the anterior maxilla. Among 25 AMs, 44.0% were of desmoplastic type, with honey-combed appearance. 84.0% of AMs were circular or oval in shape, 84.0% expanded buccally, and 36.0% invade the nasal floor. Among 89 OKCs of 88 patients, 61.8% were circular or oval, 58.4% expanded buccally, 49.4% were dentigerous, 41.6% nearly filled the maxillary sinus, and 13.5% invaded the nasal floor. 93.7% (74/79) of DCs enveloped a single tooth, and the tooth-cyst relationship was centripetal in 35, eccentric in 30, and circumferential in 9. Moreover, 98.2% (55/56) of the cysts enveloping a supernumerary tooth were DCs, while 80.9% (38/47) of the cysts enveloping the third molar were OKCs.
CONCLUSIONS
Maxillary AMs tend to grow with buccal expansion and invade the nasal floor, and DAs with honey-combed lobularity are common. Maxillary OKCs have variant shapes and tend to invaginate the maxillary sinus. The tooth-cyst relationship of dentigerous OKCs and DCs can be centripetal, eccentric, or circumferential.
Topics: Adolescent; Adult; Aged; Aged, 80 and over; Ameloblastoma; Child; Child, Preschool; Dentigerous Cyst; Female; Humans; Jaw Neoplasms; Male; Maxilla; Middle Aged; Retrospective Studies; Spiral Cone-Beam Computed Tomography; Young Adult
PubMed: 31124699
DOI: 10.1259/dmfr.20190066 -
American Journal of Veterinary Research May 2023Treatment of orofacial tumors in dogs is associated with high morbidity and reliable prognostic factors are lacking. Dynamic contrast-enhanced computed tomography...
OBJECTIVE
Treatment of orofacial tumors in dogs is associated with high morbidity and reliable prognostic factors are lacking. Dynamic contrast-enhanced computed tomography (DCECT) can be used to assess tumor perfusion. The objectives of this study were to describe the perfusion parameters of different types of orofacial tumors and to describe the changes in perfusion parameters during radiotherapy (RT) in a subset of them.
ANIMALS
11 dogs with orofacial tumors prospectively recruited.
CLINICAL PRESENTATION AND PROCEDURES
All dogs had baseline DCECT to assess blood volume (BV), blood flow (BF), and transit time (TT). Five dogs had repeat DCECT during megavoltage RT.
RESULTS
5 squamous cell carcinomas, 3 sarcomas, 1 melanoma, 1 histiocytic sarcoma, and 1 acanthomatous ameloblastoma were included. Blood volume and BF were higher in squamous cell carcinomas than in sarcomas, although no statistical analysis was performed. At repeat DCECT, 4 dogs showed a reduction in the size of their tumor during RT. Among these dogs, 3 showed an increase in BV and BF and 1 a decrease in these parameters between the baseline and the follow-up DCECT. The only dog whose tumor increased in size between the first and the second DCECT showed a decrease in BV and BF.
CLINICAL RELEVANCE
Perfusion parameters derived from DCECT were described in a series of dogs with various types of orofacial tumors. The results suggest that epithelial tumors could have higher BV and BF than mesenchymal tumors, although larger sample sizes are needed to support these preliminary findings.
Topics: Dogs; Animals; Tomography, X-Ray Computed; Carcinoma, Squamous Cell; Blood Volume; Sarcoma; Dog Diseases
PubMed: 36972698
DOI: 10.2460/ajvr.22.12.0207 -
Brazilian Dental Journal 2021The Inhibitor of Growth (ING) gene family is a group of tumor suppressor genes that play important roles in cell cycle control, senescence, DNA repair, cell...
UNLABELLED
The Inhibitor of Growth (ING) gene family is a group of tumor suppressor genes that play important roles in cell cycle control, senescence, DNA repair, cell proliferation, and apoptosis. However, inactivation and downregulation of these proteins have been related in some neoplasms. The present study aimed to evaluate the immunohistochemical profiles of ING3 and ING4 proteins in a series of benign epithelial odontogenic lesions.
METHODS
The sample comprised of 20 odontogenic keratocysts (OKC), 20 ameloblastomas (AM), and 15 adenomatoid odontogenic tumors (AOT) specimens. Nuclear and cytoplasmic immunolabeling of ING3 and ING4 were semi-quantitatively evaluated in epithelial cells of the odontogenic lesions, according to the percentage of immunolabelled cells in each case. Descriptive and statistics analysis were computed, and the p-value was set at 0.05.
RESULTS
No statistically significant differences were found in cytoplasmic and nuclear ING3 immunolabeling among the studied lesions. In contrast, AOTs presented higher cytoplasmic and nuclear ING4 labeling compared to AMs (cytoplasmic p-value = 0.01; nuclear p-value < 0.001) and OKCs (nuclear p-value = 0.007).
CONCLUSION
ING3 and ING4 protein downregulation may play an important role in the initiation and progression of more aggressive odontogenic lesions, such as AMs and OKCs.
Topics: Ameloblastoma; Cell Cycle Proteins; Cell Proliferation; Homeodomain Proteins; Humans; Odontogenic Cysts; Odontogenic Tumors; Tumor Suppressor Proteins
PubMed: 34787253
DOI: 10.1590/0103-6440202104279