-
The American Journal of Case Reports Jul 2015Ameloblastic carcinoma secondary type is an extremely rare and aggressive odontogenic neoplasm that exhibits histological features of malignancy in primary and...
BACKGROUND
Ameloblastic carcinoma secondary type is an extremely rare and aggressive odontogenic neoplasm that exhibits histological features of malignancy in primary and metastatic sites. It arises through carcinomatous de-differentiation of a pre-existing ameloblastoma or odontogenic cyst, typically following repeated treatments and recurrences of the benign precursor neoplasm. Identification of an ameloblastic carcinoma, secondary type presenting with histologic features of malignant transformation from an earlier untreated benign lesion remains a rarity. Herein, we report 1 such case.
CASE REPORT
A 66-year-old man was referred for management of a newly diagnosed ameloblastic carcinoma. He underwent radical surgical intervention comprising hemimandibulectomy, supraomohyoid neck dissection, and free-flap reconstruction. Final histologic analysis demonstrated features suggestive of carcinomatous de-differentiation for a consensus diagnosis of ameloblastic carcinoma, secondary type (de-differentiated) intraosseous.
CONCLUSIONS
Ameloblastic carcinoma, secondary type represents a rare and challenging histologic diagnosis. Radical surgical resection with adequate hard and soft tissue margins is essential for curative management of localized disease.
Topics: Aged; Ameloblastoma; Biopsy; Diagnosis, Differential; Humans; Lymphatic Metastasis; Male; Mandibular Neoplasms; Mandibular Osteotomy; Neck Dissection; Radiography, Panoramic
PubMed: 26126621
DOI: 10.12659/AJCR.893918 -
Journal of Maxillofacial and Oral... Dec 2010Ameloblastic carcinoma (AC) is a rare aggressive malignant epithelial odontogenic tumor of the maxillofacial skeleton with a distinct predilection in the mandible. It...
Ameloblastic carcinoma (AC) is a rare aggressive malignant epithelial odontogenic tumor of the maxillofacial skeleton with a distinct predilection in the mandible. It may appear de novo or originate from a pre-existing ameloblastoma or odontogenic cyst. It exhibits cytological features of ameloblastoma and carcinoma. It may present as a cystic lesion with benign clinical features or as a large tissue mass with ulceration, significant bone resorption and tooth mobility. The clinical course of ameloblastic carcinoma is typically aggressive, with extensive local destruction. Direct extension of the tumour, lymph node involvement and metastasis to various sites has been reported. Wide local excision is the treatment of choice. Regional lymph node dissection should be considered and performed selectively. Radiotherapy and chemotherapy have limited role in the treatment of ameloblastic carcinomas. Close periodic reassessment of the patient is mandatory.
PubMed: 22190836
DOI: 10.1007/s12663-010-0169-6 -
Frontiers in Oral Health 2021DNA methyltransferases (DNMTs) and the histone modification H3K9ac are epigenetic markers. This study aimed to describe the immunohistochemical expression of DNMT1,...
DNA methyltransferases (DNMTs) and the histone modification H3K9ac are epigenetic markers. This study aimed to describe the immunohistochemical expression of DNMT1, DNMT3A, DNMT3B, and H3K9ac in the dental follicle (DF), ameloblastoma (AME), and ameloblastic carcinoma (AC), correlating these expressions with the recurrence and aggressive behavior in ameloblastoma. Immunohistochemical reactions were performed in 10 human DFs, 38 ameloblastomas, and 6 AC samples. Another 59 ameloblastomas assembled in a tissue microarray were used to compare the immunoexpression with the clinical, radiographic, and histopathological characteristics and the presence of BRAFv600e mutation. Each slide was digitized as a high-resolution image and quantified by Aperio ScanScope Nuclear V9 software. All statistical analyzes were performed using GraphPad Prism statistical software. DNMT3B expression was higher in ameloblastomas than in the DFs, while the AC overexpressed all proteins. The ameloblastomas with BRAFv600e mutation, vestibular/lingual, or vestibular/palatine bone cortical disruption and maxilla involvement showed DNMT1 overexpression, while recurrent cases had high DNMT3B levels. DNA methylation and histone modification might play a role in the development, clinical aggressiveness, and recurrence rates of ameloblastoma, such as the progression to AC. Further investigation about gene methylations in ameloblastomas is needed to better understand its relationship with aggressiveness and recurrence.
PubMed: 35048062
DOI: 10.3389/froh.2021.751162 -
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 -
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 -
Journal of Veterinary Diagnostic... May 2022Ameloblastic carcinoma is a malignant odontogenic neoplasm that has been reported only rarely in veterinary species. A 16-y-old Arabian crossbred mare was presented for... (Review)
Review
Ameloblastic carcinoma is a malignant odontogenic neoplasm that has been reported only rarely in veterinary species. A 16-y-old Arabian crossbred mare was presented for evaluation of a hard mass on the body of the mandible, with evidence of osteolysis on radiographs. Incisional biopsies revealed an invasive neoplasm comprised of spindloid epithelial cells with a high mitotic count and partial dual cytokeratin-vimentin immunoreactivity. The horse was euthanized because of rapid tumor progression 3 mo after presentation. Postmortem evaluation revealed partial obliteration of the mandible by a large, firm-to-hard, tan, locally destructive and invasive mass with no gross or histologic evidence of metastasis. Postmortem histology revealed a poorly differentiated epithelial neoplasm with variably prominent features suggestive of odontogenic histogenesis: a plexiform ribbon architecture, infrequent basilar palisading with antibasilar nuclei, rare basilar cytoplasmic clearing, subepithelial matrix hyalinization, and partial dual cytokeratin-vimentin immunoreactivity. Features of malignancy included regions of necrosis, pronounced cellular atypia, a high mitotic count, extensive tissue invasion and local tissue destruction, and extension of neoplastic cells beyond the margins of the mandibular bone. Collectively, these features are most consistent with mandibular ameloblastic carcinoma. Including our case described here, ameloblastic carcinoma has been reported in only 5 horses. The microscopic features reported most consistently are dual cytokeratin-vimentin immunoreactivity, a high mitotic count, and basilar palisading. Ameloblastic carcinoma should be considered as a differential diagnosis for rapidly growing, locally invasive masses arising from the dentate jaw of horses.
Topics: Ameloblastoma; Animals; Carcinoma; Female; Horse Diseases; Horses; Keratins; Mandibular Neoplasms; Odontogenic Tumors; Vimentin
PubMed: 35037545
DOI: 10.1177/10406387211068459 -
Orphanet Journal of Rare Diseases Nov 2020Ameloblastic carcinoma (AC) is an odontogenic malignant tumor which is closely related to benign ameloblastoma. Because of its rarity, diagnosis and treatment are...
BACKGROUND
Ameloblastic carcinoma (AC) is an odontogenic malignant tumor which is closely related to benign ameloblastoma. Because of its rarity, diagnosis and treatment are difficult. In this study, we summarized and analyzed the clinical and biological characteristics of AC.
RESULTS
Fifteen patients with AC and a median age of 53 years were identified. Among of them, five patients who were tested carried a BRAF-V600E mutation. Two patients presented with cervical lymph nodes and lung metastases. Primary AC was more invasive, and the bone destruction ability of the primary type was more radical than that of the secondary type.
CONCLUSIONS
This study revealed that the BRAF-V600E mutation was related to the aggressive behavior of AC, and early radical resection is crucial. Moreover, targeted therapy may be a new direction in the future.
Topics: Ameloblastoma; Carcinoma; Humans; Middle Aged; Odontogenic Tumors
PubMed: 33176823
DOI: 10.1186/s13023-020-01603-5 -
The Pan African Medical Journal 2018Ameloblastic carcinoma is a rare malignant odontogenic neoplasm that exhibits histological features of ameloblastoma in combination with cytological atypia. It may arise... (Review)
Review
INTRODUCTION
Ameloblastic carcinoma is a rare malignant odontogenic neoplasm that exhibits histological features of ameloblastoma in combination with cytological atypia. It may arise de novo or secondarily through malignant de-differentiation of pre-existing ameloblastoma or odontogenic cyst. Secondary ameloblastic carcinomas often results from repeated surgical intervention, which is a mainstay of odontogenic tumor management in resource limited settings. To date, relatively few cases of ameloblastic carcinomas have been reported and many cases have been misdiagnosed as ameloblastoma. This is due to its wide range of clinicopathological feature which range from indolent to aggressive. It may present as an aggressive ulcerated mass or as a simple cystic lesion; hence, it often challenging to delineate from its benign counterpart, ameloblastoma.
METHODS
this study reviewed the clinicopathological data on 157 cases of odontogenic tumors diagnosed over a 10 years period from the pathology archive of the Oral Pathology Unit of Obafemi Awolowo University Teaching Hospital Complex (OAUTHC), Ile-Ife, Nigeria.
RESULTS
of all these cases, we identified that 64.9% were Ameloblastomas, while 8.3% were ameloblastic carcinomas. Primary subtypes of ameloblastic carcinoma constituted 23.08%, while 69.23% of the cases were of the secondary subtype. We also found that the secondary subtype of ameloblastic carcinomas showed a higher mean duration value of 7.7 years. Most lesions were found in posterior mandible and presented with ulceration, perforation and ill-defined borders radiographically.
CONCLUSION
this study is among the few that have documented higher frequency of secondary ameloblastic carcinoma in the scientific literature.
Topics: Adolescent; Adult; Ameloblastoma; Child, Preschool; Female; Hospitals, Teaching; Humans; Male; Mandibular Neoplasms; Middle Aged; Nigeria; Odontogenic Cysts; Odontogenic Tumors; Retrospective Studies; Young Adult
PubMed: 31497184
DOI: 10.11604/pamj.2018.31.208.14660 -
National Journal of Maxillofacial... 2018Ameloblastic carcinoma is considered to be a rare epithelial malignant neoplasm of odontogenic origin occurring mainly in the mandible. Ameloblastic carcinoma has been a... (Review)
Review
Ameloblastic carcinoma is considered to be a rare epithelial malignant neoplasm of odontogenic origin occurring mainly in the mandible. Ameloblastic carcinoma has been a topic of controversy regarding management from past many years. We reviewed 86 cases of mandibular ameloblastic carcinoma from 1981 to 2014, on the basis of the electronic search of peer-reviewed journals in MEDLINE (PubMed) database. Age, sex, tumor size, treatment delivered, recurrence, metastasis, follow-up period, and dead/alive status are tabulated, and the data are analyzed. The mean age was 43.47 years with standard deviation ± 21.09. The age range was between 15 and 91 years, and male to female ratio was 2.18:1. Knowledge gained from the present review would help in establishing the best therapeutic options for ameloblastic carcinoma, and it also encourages the further reporting of ameloblastic carcinoma.
PubMed: 29937652
DOI: 10.4103/njms.NJMS_33_16