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International Journal of Environmental... Apr 2021Ameloblastic carcinoma is a rare malignant odontogenic neoplasm with a poor prognosis. It can arise de novo or from a pre-existing ameloblastoma. Research into stemness...
Ameloblastic carcinoma is a rare malignant odontogenic neoplasm with a poor prognosis. It can arise de novo or from a pre-existing ameloblastoma. Research into stemness marker expression in ameloblastic tumours is lacking. This study aimed to explore the immunohistochemical expression of stemness markers nestin, CD138, and alpha-smooth muscle actin (alpha-SMA) for the characterisation of ameloblastic tumours. Six cases of ameloblastoma and four cases of ameloblastic carcinoma were assessed, including one case of ameloblastic carcinoma arising from desmoplastic ameloblastoma. In all tumour samples, CD138 was positive, whilst alpha-SMA was negative. Nestin was negative in all but one tumour sample. Conversely, the presence or absence of these markers varied in stroma samples. Nestin was observed in one ameloblastic carcinoma stroma sample, whilst CD138 was positive in one ameloblastoma case, one desmoplastic ameloblastoma case, and in two ameloblastic carcinoma stroma samples. Finally, alpha-SMA was found positive only in the desmoplastic ameloblastoma stroma sample. Our results suggest nestin expression to be an indicator for ameloblastic carcinoma, and CD138 and alpha-SMA to be promising biomarkers for the malignant transformation of ameloblastoma. Our data showed that nestin, CD138, and alpha-SMA are novel biomarkers for a better understanding of the origins and behaviour of ameloblastic tumours.
Topics: Actins; Ameloblastoma; Biomarkers; Humans; Mandibular Neoplasms; Nestin
PubMed: 33917771
DOI: 10.3390/ijerph18083899 -
SpringerPlus 2015Ameloblastoma is a benign odontogenic neoplasm of the jaw, rarely presenting as a malignant tumor. Although it is very important to discriminate ameloblastoma from...
INTRODUCTION
Ameloblastoma is a benign odontogenic neoplasm of the jaw, rarely presenting as a malignant tumor. Although it is very important to discriminate ameloblastoma from ameloblastic carcinoma in order to decide the appropriate operative procedure, this is difficult using conventional CT and MRI.
CASE DESCRIPTIONS
We report a case of maxillar ameloblastoma in a 78-year-old man where FDG-PET/CT was useful for making this discrimination. CT demonstrated a 31 × 43 × 46-mm mass in the left posterior maxillary sinus with destruction of its posterior and lateral wall and alveolar bone. MRI demonstrated a hypo- to isointense heterogeneous pattern on T1WI, heterogeneous hyperintensity with a prominent high-signal spot on T2WI, high signal intensity on DWI reflecting restricted diffusion, and strong heterogeneous enhancement. Because FDG-PET/CT showed mild FDG uptake (SUVmax 2.40) by the mass, ameloblastoma, rather than ameloblastic carcinoma, was considered to be the correct diagnosis.
DISCUSSION AND EVALUATION
It appears that ameloblastic carcinoma shows intense FDG uptake, whereas ameloblastoma shows mild or moderate FDG uptake, and only rarely intense FDG uptake. Our experience suggests that FDG-PET/CT may be effective for discriminating ameloblastoma from ameloblastic carcinoma. Especially, in cases showing mild FDG uptake, benign ameloblastoma would seem the most likely diagnosis.
CONCLUSIONS
FDG-PET/CT may be useful as an adjunctive modality for diagnosis, treatment planning and surveillance of ameloblastoma and ameloblastic carcinoma.
PubMed: 26101729
DOI: 10.1186/s40064-015-0998-3 -
Journal of Neurosurgery. Case Lessons Sep 2023Ameloblastic carcinoma (AC) is a rare odontogenic carcinoma with histological features resembling ameloblastoma. Metastasis to distant organs and direct expansion into...
BACKGROUND
Ameloblastic carcinoma (AC) is a rare odontogenic carcinoma with histological features resembling ameloblastoma. Metastasis to distant organs and direct expansion into the skull base structures are associated with a poor clinical outcome. This rare case of AC metastasis to the pituitary gland presented without local recurrence at the primary focus of the maxilla.
OBSERVATIONS
A 47-year-old man had a 2-year history of AC in the right maxilla. Computed tomography for his regular checkup incidentally demonstrated pituitary tumor, rapidly growing over 2 months. He presented with the recent onset of panhypopituitarism and visual field defect. Magnetic resonance imaging showed a large, irregularly shaped intrasellar and suprasellar lesion with chiasmal compression. Endoscopic endonasal transsphenoidal surgery was performed for decompression of the optic apparatus to avoid intracranial spread. Histopathology confirmed metastatic AC, and a genetic panel test confirmed BRAF V600E mutation. Stereotactic radiotherapy (SRT) with the CyberKnife system was administered to the residual tumor. Remarkable tumor shrinkage was obtained, and panhypopituitarism was resolved 12 months later.
LESSONS
A multidisciplinary treatment strategy including maximal safe resection to avoid dissemination in combination with SRT may be crucial for local control with the preservation of pituitary and visual functions in patients with solitary pituitary metastatic AC.
PubMed: 37728240
DOI: 10.3171/CASE23264 -
Ear, Nose, & Throat Journal Apr 2024Ameloblastic carcinoma (AC) represents a distinct challenge in the realm of odontogenic malignancies due to its rarity and aggressive nature. We present a unique case of...
Ameloblastic carcinoma (AC) represents a distinct challenge in the realm of odontogenic malignancies due to its rarity and aggressive nature. We present a unique case of AC in a 70-year-old male, retired dry cleaner, with symptoms initially suggestive of chronic allergic rhinitis and recurrent acute sinusitis with asymmetric facial edema and paresthesia. Detailed evaluation revealed a prominent mass in the right maxillary sinus with extensive cortical destruction. Pathological assessment post-right maxillectomy identified a high-grade AC with malignant spindle cell transformation. The patient underwent subsequent interventions, including neck dissection and radiation therapy. Twelve months post-presentation, the patient was recovering appropriately without evidence of recurrence of malignancy. This case highlights the diagnostic challenges posed by AC as well as its unique presentations emphasizing the importance of a comprehensive approach and multidisciplinary management. It also raises considerations about potential chemical exposure implications in AC development.
PubMed: 38561954
DOI: 10.1177/01455613241245207 -
Oncology Letters Dec 2016Ameloblastic carcinoma (AC) is an uncommon malignant odontogenic tumor that can be difficult to differentiate from ameloblastoma and can arise directly as an...
Ameloblastic carcinoma (AC) is an uncommon malignant odontogenic tumor that can be difficult to differentiate from ameloblastoma and can arise directly as an undifferentiated lesion or from a pre-existing benign lesion. The current study presents a novel case of primary maxillary AC and review the literature on AC of the maxilla. The review of the literature indicates that secondary tumors and posterior localization are associated with a higher tendency for recurrence and, often, multiple recurrences. Surgical therapy, eventually followed by radiotherapy, is the treatment modality most frequently applied, while the role of chemotherapy remains unclear. Several new cases of maxillary AC have been recently described in literature, making this pathology more frequent than previously considered; this is perhaps an indication of an increased diagnostic sensibility, rather than a real increase in the incidence of the disease itself.
PubMed: 28105148
DOI: 10.3892/ol.2016.5272 -
Oncology Letters Aug 2014The diagnosis of ameloblastic carcinoma is often difficult and the optimal treatment methods remain controversial. The current study retrospectively investigated the...
The diagnosis of ameloblastic carcinoma is often difficult and the optimal treatment methods remain controversial. The current study retrospectively investigated the optimal diagnosis and treatment methods of 12 ameloblastic carcinoma patients at the West China Hospital of Stomatology, Sichuan University (Chengdu, China), and 20 patients selected from the PubMed database, were reviewed. The clinical features, diagnosis and outcome of the different treatments were evaluated. Ameloblastic carcinoma occurred in 12 out of a total of 538 ameloblastoma patients; the majority were of the primary type. Of the 538 ameloblastoma patients, 294 were male, 244 were female with a male to female ratio of 1.2:1. The predilection age is 20-30 years, which accounts for 40% of the total. In total, 461 cases were in the mandible and 77 were located in the maxilla. The cure rate of the primary type and the recurrence rate of the secondary type tumors were higher in the patients from the West China Hospital of Stomatology compared with those reported in the literature. In particular, a case with a long-term survival of 30 years is presented, which is considered to be relatively rare. The evolution of the clinical course has experienced three stages: Ameloblastoma (1978) followed by metastatic ameloblastoma (2000) and finally ameloblastic carcinoma (2008). To avoid recurrence, wide local excision with postoperative radiation therapy is required. While novel therapeutic regimens should also be considered as appropriate, including carbon ion therapy and Gamma Knife stereotactic radiosurgery. However, controlled studies with larger groups of patients are required to increase the accuracy of results.
PubMed: 25013517
DOI: 10.3892/ol.2014.2230 -
Journal of Oral and Maxillofacial... 2023Ameloblastic carcinoma (AC) is a rare, primary epithelial odontogenic malignant neoplasm. It is the malignant counterpart of ameloblastoma. It comprises 1% of all cysts...
Ameloblastic carcinoma (AC) is a rare, primary epithelial odontogenic malignant neoplasm. It is the malignant counterpart of ameloblastoma. It comprises 1% of all cysts and tumours occurring in the jaws, arising from tissues associated with odontogenic epithelium. The objective of the present study was to describe a clinical case of a 63-year-old male with an enlargement in the mandible on the left side. Panoramic radiography revealed a radiolucent area with poorly defined borders, and an incisional biopsy was performed for the histopathological study using immunomarkers such as SOX2 and Ki-67. Ki-67 is considered a marker of cell proliferation, and SOX2 reportedly participates in the development of the ameloblastic epithelium lineage and is associated with a more aggressive clinical course. A final histopathological diagnosis of AC was given. Unfortunately, the patient died one week before surgical resection (the surgical treatment of choice for AC).
PubMed: 37234302
DOI: 10.4103/jomfp.jomfp_339_22 -
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 -
BMC Oral Health Mar 2021Malignant odontogenic tumours are extremely rare tumours occurring within the jaws. Our study was performed to determine the demographic and clinico-pathological...
BACKGROUND
Malignant odontogenic tumours are extremely rare tumours occurring within the jaws. Our study was performed to determine the demographic and clinico-pathological features of malignant odontogenic tumours amongst a multi-ethnic Asian population.
METHODS
This was a retrospective cross-sectional study of malignant odontogenic tumours diagnosed at the Institute for Medical Research, Malaysia, from 2009 to 2019. All cases were independently reviewed and reclassified following the criteria set out in the latest edition of the World Health Organization 2017 reference text. Demographic and clinico-pathological data were recorded for each case.
RESULTS
Twenty-four cases of malignant odontogenic tumours were identified. The patients' age ranged from 16 to 79 years with the mean age at diagnosis being 50.8 years (SD = 16.18). There was a male predominance (66.7%) in this cohort of patients. The ethnic distribution appeared to reflect the Malaysian population with most cases seen amongst the Malay ethnic group (66.7%). Ameloblastic carcinoma was the most frequently diagnosed malignant odontogenic tumour (45.8%) and was also predominantly seen in males (90.9%). All patients with clear cell odontogenic carcinoma were females. There was no obvious sex predilection in primary odontogenic carcinoma not otherwise specified (NOS). The mandible (79.2%) was more frequently involved compared to the maxilla.
CONCLUSIONS
Diagnosis and management of malignant odontogenic tumours are challenging due to the rarity of these tumours. Our study has elucidated the clinico-pathological features of malignant odontogenic tumours seen in a multi-ethnic Asian population.
Topics: Adolescent; Adult; Aged; Cross-Sectional Studies; Female; Humans; Malaysia; Male; Middle Aged; Odontogenic Tumors; Referral and Consultation; Retrospective Studies; Young Adult
PubMed: 33736630
DOI: 10.1186/s12903-020-01365-3 -
Ear, Nose, & Throat Journal May 2023Ameloblastic carcinoma (AC) is a rare and aggressive malignant epithelial odontogenic tumor making up less than 1% of malignant head and neck tumors. The majority of...
Ameloblastic carcinoma (AC) is a rare and aggressive malignant epithelial odontogenic tumor making up less than 1% of malignant head and neck tumors. The majority of cases occur in the mandible with a minority occurring in the maxilla. Most occur de novo, while rare cases of AC have resulted from transformation from ameloblastoma. Here, we present a case in which a 30-year-old man presented with proptosis and a recurrent right temporal mass, which had been previously diagnosed as ameloblastoma on surgical pathology. CT findings demonstrated local invasion, and he was subsequently taken to the operating room for right craniotomy, infratemporal and middle cranial fossa tumor resection, and right modified radical neck dissection with reconstruction. Final pathology, which included areas of early focal necrosis, loss of peripheral palisading, and hyperchromatism, confirmed the diagnosis of ameloblastoma with transformation to AC. We further discuss radiologic and histopathological signs of this rare tumor, as well as recommended treatment modalities.
PubMed: 37158333
DOI: 10.1177/01455613231172857