Ameloblastic Fibro-odontosarcoma Research Articles

Ameloblastic fibro-odontosarcoma with mandibular bone invasion and regional lymph node metastasis in a cat: case report

Odontogenic tumours are uncommon neoplasms in domestic animals, mostly solitary and locally infiltrative, but rarely metastatic. We report the case of a 13-year-old neutered male cat presented with a mandibular gingival neoformation. A computed tomography scan revealed an irregular neoformation with marked post-contrast enhancement, associated with lysis of the incisive bone and mandibular symphysis. Histologically, the oral mucosa and mandibular bone were infiltrated by a neoplasm consisting of a mixed population of odontogenic epithelium admixed with bundles of odontogenic ectomesenchyme, multifocally associated with hard tissue deposition. A spindloid cell component had metastasized to the right mandibular lymph node. The epithelial component was immunoreactive for cytokeratins (CK) 5/6, CK 14, pancytokeratin (CK AE1/AE3) and p63; the ectomesenchymal component was vimentin positive. A final diagnosis of ameloblastic fibro-odontosarcoma with bone invasion and lymph node metastasis was made. The findings indicate the metastatic potential of this rare tumour.

Clinical management of primary odontogenic sarcoma in the mandible: a case report after WHO nomination

Ameloblastic fibro-odontosarcoma (AFOS) now designated as odontogenic sarcoma is an extremely rare odontogenic tumor, which histologically presents as a biphasic neoplasm with a malignant mesenchymal component plus ameloblastic epithelium. Here we report a 27-year-old Chinese female with the complaint of a painful swelling for half a month in the right mandible. A segmental mandibulectomy, with an immediate mandibular reconstruction using a free vascularized osteocutaneous fibular flap was performed using surgical guide models. Histological analysis revealed a primary odontogenic sarcoma. The postoperative period was uneventful, and no clinical indication of recurrence or metastasis was observed during the 3-year follow-up. No adjuvant therapy was proposed. This is the first odontogenic sarcoma case reported in China after the new World Health Organization classification of odontogenic lesions.

Mixed odontogenic tumors: A review of the clinicopathological and molecular features and changes in the WHO classification.

BACKGROUNDAmeloblastic fibromas and ameloblastic fibrosarcomas are rare odontogenic tumors, and controversy exists in the classification of cases presenting hard-tissue production: Ameloblastic fibrodentinoma (AFD) and ameloblastic fibro-odontoma (AFO). These cases are currently considered “developing odontomas” (hamartomatous lesions). AIMTo analyze the clinicopathologic features of these lesions and discuss the changes in the 2017 World Health Organization classification.METHODSAn electronic literature search was performed in the PubMed/MEDLINE database. An electronic search of the English language literature was performed and last updated in September 2020 in the PubMed/MEDLINE database using the following terms: “ameloblastic fibroma”, “ameloblastic fibrodentinoma”, “ameloblastic fibro-odontoma”, “ameloblastic sarcoma”, “ameloblastic fibrosarcoma”, “ameloblastic fibrodentinosarcoma”, “ameloblastic fibroodontosarcoma” and “odontogenic carcinosarcoma”. The inclusion criteria were odontogenic tumor series, case reports and systematic reviews that provided sufficient clinical, radiological and microscopic documentation to confirm the diagnosis. RESULTSThe database search strategy resulted in 947 papers. Articles focusing on other topics, articles that were not in English, duplicate articles, and articles without fulfilling the inclusion criteria were excluded. Finally, 96 publications were included in this review to describe and discuss the main features of the searched entities. Several aspects of AFO and AFD, such as biological behavior, age of occurrence, amount of hard tissue, and potential for malignant transformation into odontogenic sarcomas, support the neoplastic nature in most of the reported cases. Considering the clinical, radiographic, histopathological and molecular characteristics of odontogenic lesions with hard tissue production, we suggest that these types of lesions should continue to be recognized as odontogenic tumors by maintaining the classically used terms. CONCLUSIONThis recommendation will be relevant for future clinical, microscopic, and molecular studies to better understand the biology of these interesting odontogenic tumors.

Ameloblastic fibrodentinosarcoma and ameloblastic fibro-odontosarcoma: A systematic review

The purpose of the present review was to integrate the available data published on ameloblastic fibrodentinosarcoma (AFDS) and ameloblastic fibro-odontosarcoma (AFOS) into a comprehensive analysis of their clinical/radiologic features. An electronic search was undertaken in July/2017. Eligibility criteria included publications having enough clinical/radiological/histological information to confirm the diagnosis. Seventeen publications (8 AFDS, 9 AFOS) were included. The patients with the two different lesions had a similar mean age. The lesions were both more prevalent in mandibles than in maxillae, all showed bone expansion and similar rates of cortical bone perforation, tooth displacement, and locular appearance at radiological exams. Mean follow-up time was higher for AFDS, and the mean size of the lesions were larger for AFDS, although without a statistically significant difference. None of AFDS recurred, while 2 AFOS recurred. As only few cases of AFDS and AFOS have been reported, additional reports are necessary to add evidence to the study of clinical and radiologic features of these lesions.

Ameloblastic fibro-odontosarcoma of the mandible with active epithelial proliferation: A rare case report.

Ameloblastic fibro-odontosarcoma (AFOS) is an extremely rare subtype of odontogenic sarcoma, with no more than 19 cases reported in the English literature to date. AFOS is a biphasic neoplasm, with deposits of dentin and enamel matrix. We herein present a case of AFOS with active epithelial proliferation in a 31-year-old female patient. The patient was referred to the West China Hospital of Stomatology (Chengdu, China) due to a 6-month history of a swelling in the left mandible. Following clinical and radiological examination, the initial preoperative diagnosis was ameloblastoma, with local invasion and the possibility of malignant transformation. Left hemimandibular resection was subsequently performed. The postoperative histopathological diagnosis was AFOS, accompanied by active epithelial proliferation. Immunohistochemically, cytokeratin (CK)14 and CK19 were intensely positive in the epithelium, whereas the mesenchymal cells were strongly positive for vimentin. The Ki-67 labeling index was considerably higher in the mesenchymal component (mean, 40%) compared with that in the epithelial element (mean, 5–8%). Three months after the surgical procedure, the patient remained clinically and radiologically disease-free.

Ameloblastic fibro-odontosarcoma of the mandible in a pediatric patient

IntroductionAmeloblastic fibro-odontosarcoma is an extremely rare subtype of odontogenic sarcoma, with only 13 cases reported in the literature. Case reportA 4-year-old male presented with a painless mandibular swelling, which appeared 4months previously. Cone beam computed tomography revealed an extensive, ill-circumscribed, multilocular radiolucency of the right mandible extending from the first deciduous molar to the ramus with slightly dense opacities. Histological examination of the incisional biopsy specimen revealed a biphasic tumor with sarcomatous mesenchyme and benign ameloblastic epithelial component compatible with a diagnosis of ameloblastic fibrosarcoma. A right hemimandibular resection was performed. Areas of deposition of dentinoid and enamel material closely adjacent to ameloblastic epithelium were noted in the excised specimen. A final diagnosis of ameloblastic fibro-odontosarcoma was made. After four years of close follow-up, there is no sign of recurrence or metastasis. ConclusionAlthough rare, ameloblastic fibro-odontosarcoma should be considered in the differential diagnosis of jaw lesions with radiographic radiolucencies exhibiting poorly circumscribed outlines and containing radiopaque material. Definite diagnosis depends on histopathological examination. Complete surgical excision is the treatment of choice.

Pediatric Ameloblastic Fibro-Odontosarcoma of the Mandible: A Challenge of Diagnosis and Treatment

Pediatric Ameloblastic Fibro-Odontosarcoma of the Mandible: A Challenge of Diagnosis and Treatment

Ameloblastic Fibrosarcoma: Report of a Case, Study of Immunophenotype, and Comprehensive Review of the Literature

Ameloblastic fibrosarcoma (AFS) is a rare odontogenic neoplasm exhibiting a benign ameloblastomatous epithelial component admixed within a sarcomatous mesenchyme. It is considered the malignant counterpart of ameloblastic fibroma (AF) and can arise de novo or from transformed AF.1 AFS is a locally aggressive tumor that seldom metastasizes. According to the World Health Organization classification, AFS is considered an odontogenic sarcoma. Other entities that fall under this category include ameloblastic fibrodentinosarcoma (AFDS), ameloblastic fibro-odontosarcoma (AFOS), and odontogenic carcinosarcoma (OCS).

Ameloblastic fibro-odontosarcoma of the mandible: imaging findings

We report a histologically verified case of ameloblastic fibro-odontosarcoma (AFOS) affecting the right mandible. CT scanning showed an expansile intraosseous mass with ill-defined borders and slightly dense opacities.

Ameloblastic fibro-odontosarcoma: a case report

This paper reports one case, of an ameloblastic fibro-odontosarcoma (AFOS) affecting the mandible, in a 12-year-old girl. This neoplasm is a rare odontogenic neoplasm. To the authors’ knowledge this is the fifteenth case of AFOS reported in English. The patient's chief complaint was a swelling in the face for 6 months. An incisional biopsy was performed diagnosing the case as an ameloblastic fibroma. After radiography ameloblastic fibro-odontoma was diagnosed. Computed tomography was performed and a stereolithography model made to plan the surgical procedures. A hemimandibulectomy followed by a vascularized fibular flap was then proposed. The surgery was uneventful. Microscopic features diagnosed an AFOS. After 23 months of close follow-up there is no sign of recurrence or metastasis. Dental implants were recently placed in the fibular flap.

Differential Expression of Collagen IV alpha1 to alpha6 Chains in Basement Membranes of Malignant Odontogenic Tumors

Type IV collagen, the major component of basement membrane (BM), demonstrates a stage- and position-specific distribution of its isoforms during tooth development. To determine its localization in BM of malignant odontogenic neoplasms, immunohistochemistry using six anti-alpha(IV) chain-specific monoclonal antibodies was performed. Results disclosed that BM demonstrated an irregular alpha(IV) chain profile in tooth germ as compared to benign odontogenic neoplasms. No alpha3(IV) chains were detected. Expression of alpha1(IV)/alpha2(IV) and alpha4(IV), alpha5(IV)/alpha6(IV) chains was ameloblastic fibro-odontosarcoma, but not the inductive hard tissues. Ameloblastic carcinoma showed specific alpha1(IV)/alpha2(IV) chain loss, while primary intraosseous carcinoma demonstrated a discontinuous alpha1(IV)/alpha2(IV) and alpha5(IV)/alpha6(IV) staining pattern. The present results suggest that modification and remodeling of BM collagen IV alpha chains occur during malignant odontogenic neoplasms' progression.

Differential expression of collagen IV α1 to α6 chains in basement membranes of benign and malignant odontogenic tumors

Type IV collagen, the major component of basement membrane (BM), demonstrates a stage- and position-specific distribution of its isoforms during tooth development. To determine its localization in BM of odontogenic neoplasms, immunohistochemistry using six anti-alpha(IV) chain-specific monoclonal antibodies was performed. Results disclosed that BM demonstrated an irregular alpha(IV) chain profile in malignant odontogenic tumors as compared to benign odontogenic neoplasms. No alpha3(IV) chains were detected. Expression of alpha1(IV)/alpha2(IV) and alpha5(IV)/alpha6(IV) chains was stronger in desmoplastic ( n=3) than in ordinary (n=5) ameloblastomas. The adenomatoid odontogenic tumor ( n=2) distinctly expressed these chains in BM of cribriform areas and hyaline materials (which was also alpha4(IV)-positive), but weakly around epithelial whorls/rosettes/nests and mineralized foci. These five chains also stained BM and tumor cells of ameloblastic fibroma ( n=3) and ameloblastic fibro-odontosarcoma (n=1), but not the inductive hard tissues. Ameloblastic carcinoma ( n=2) showed specific alpha1(IV)/alpha2(IV) chain loss, while primary intraosseous carcinoma ( n=1) demonstrated a discontinuous alpha1(IV)/alpha2(IV) and alpha5(IV)/alpha6(IV) staining pattern. The present results suggest that modification and remodeling of BM collagen IValpha chains occur during odontogenic neoplasms' progression.

Ameloblastie fibromas and related tumors in cattle

This article concerns rare odontogenic tumors that occur predominantly in the mandibular incisor region of young cattle and which have often in the past been referred to as ameloblastomas, or as the outdated synonym, adamantinoma. Twenty-two examples from the literature and two new ones were studied. Six consisted of epithelial islands which resembled those of ameloblastoma but which were located within a cellular fibrous connective tissue that was the second component of the tumor; these mixed odontogenic tumors therefore represented ameloblastic fibromas, not ameloblastomas. Eight consisted of a combination of ameloblastic fibroma and odontoma and therefore were ameloblastic fibro-odontomas, and one was apparently malignant (ameloblastic fibro-odontosarcoma). Excluding this last lesion, these tumors should respond well to enucleation, like their human counterparts but, to confirm this hypothesis, the margins of future examples should be carefully examined to determine that they are well-demarcated, not invasive. The microscopic features of the remaining 9 tumours could not be evaluated adequately, while another 17 tumors in cattle and water buffalo reported briefly could not be studied to any extent because of insufficient information.

Ameloblastic Odontosarcoma (Ameloblastic Fibro‐odontosarcoma) in the Mandible

A case of ameloblastic odontosarcoma (ameloblastic fibro-odontosarcoma) originating in the mandible is reported. The patient was a 23-year-old Japanese male with diffuse swelling of the mandible from the left premolar to the ramus region. A biopsy specimen showed the histopathological features of ameloblastic fibrosarcoma. Histologic examination of the surgical specimen revealed various amounts of dysplastic dentin and dentinal matrix which were closely associated with both mesenchymal and epithelial components throughout the lesion. In addition, a small amount of abortive enamel matrix, which was in close contact with ameloblastoid cells of epithelial nests, was found by extensive sampling of the resected specimen. Therefore the present case was diagnosed as ameloblastic odontosarcoma. After 2 years, the patient died of uncontrollable local recurrence and extension to the cranial base.