ABSTRACT
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.
Subject(s)
Ameloblastoma , Carcinoma , Mandibular Neoplasms , Odontogenic Tumors , Ameloblastoma/pathology , Carcinoma/pathology , Humans , Mandible/pathology , Mandibular Neoplasms/pathology , Odontogenic Tumors/pathologyABSTRACT
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.
Subject(s)
Ameloblastoma , Odontogenic Tumors , Ameloblastoma/diagnosis , Ameloblastoma/epidemiology , Cell Transformation, Neoplastic , Humans , Neoplasm Recurrence, Local/diagnosis , Neoplasm Recurrence, Local/epidemiology , PrognosisABSTRACT
Metastasizing ameloblastoma (MA) is a very rare odontogenic tumor with 2% of incidence rate. It exhibits benign histopathological features and malignant intrinsic quality in the form of metastasis which makes it a little more than a pathological curiosity. Various molecular aspects related with malignant behavior have been discussed. Because of this, it provides a diagnostic challenge for clinicians and surgeons. It is an elusive lesion which should be more researched and studied so that definitive diagnostic features can be put forward. The objective of this paper is to review the molecular aspect involved in the pathogenesis of MA which will aid in differentiating non-MA from MA and thus helping in providing proper treatment at an early stage.
Subject(s)
Ameloblastoma/etiology , Ameloblastoma/metabolism , Ameloblastoma/diagnosis , Ameloblastoma/therapy , Biomarkers, Tumor , Disease Management , Disease Susceptibility , Epithelial-Mesenchymal Transition , Humans , Neoplasm Metastasis , Neoplasm Staging , Tumor MicroenvironmentABSTRACT
Ameloblastoma is a locally aggressive, epithelial odontogenic tumor involving mandibles and maxillas. Distant metastasis is a very rare condition and is designated as metastasizing (malignant) ameloblastoma despite its benign histological appearance. Up to now, only 27 well-documented cases of metastasizing ameloblastomas are reported in the literature, and lung is the most commonly involved organ. In previous reports of pulmonary metastasizing ameloblastomas, there was little description of the histopathologic finding. Here, the authors report 2 cases of pulmonary metastasizing ameloblastomas with special emphasis on their interesting, interstitial spread along alveolar septa, resulting in a unique 2-cell pattern under microscopic examination. Pulmonary metastasizing ameloblastoma may pose difficulty in diagnosis if the pathologist is not aware of patient's clinical history of ameloblastoma.