Ameloblastic fibro-odontosarcoma: a case report

doi:10.1016/j.ijom.2008.11.025

International Journal of Oral and Maxillofacial Surgery

Volume 38, Issue 3, March 2009, Pages 289-292

https://doi.org/10.1016/j.ijom.2008.11.025 Get rights and content

Abstract

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.

Section snippets

Case report

A 12-year-old girl was referred to the Oral and Maxillofacial Surgery Service at Hospital Nove de Julho (Instituto Oncológico), Juiz de Fora (MG), Brazil, in June 2006, with a left facial swelling and a complaint of 6 months’ mild pain due to masticatory trauma in the gum. Oral examination revealed a fixed and protruding mass, tender to palpation and covered with a whitish and red mucosa and pseudo-membrane (Fig. 1). Orthopantomography revealed a poorly defined radiolucency extending from the

Discussion

AFS are rare, true, mixed neoplasms exhibiting a benign epithelial component and malign ectomesenchymal cells2, 3, 6, 7, 9. Some authors consider that AFS are the malignant counterparts of the AF1, 3, 6, 9. AFO are considered possible precursor lesions for AFDS and AFOS². Carlos et al.² state that, 64 cases of AFS and 14 cases of AFDS/ AFOS have been reported in the English-language literature, up to 2005. To the authors’ knowledge, the case presented here is the fifteenth case of AFDS/AFOS

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There are more references available in the full text version of this article.

Cited by (20)

A case of a huge mandibular tumor composed of ameloblastoma and high-grade sarcoma
2019, Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology
Ameloblastoma is a benign odontogenic tumor of the jaw. Sarcoma is a rare neoplasm that accounts for only about 1% of all tumors. Here, we report a quite rare case of simultaneous occurrence of an ameloblastoma and a high-grade sarcoma in the mandible of a 65-year-old man. He was referred to our hospital because of his difficulties in eating and drinking due to a remarkable swelling of the right mandible. He had first noticed the swelling about six years before and consulted a clinic one year before, but he had refused any examination or treatment. He underwent an incisional biopsy, and it revealed a high-grade spindle cell sarcoma with massive necrosis. Therefore, we suggested surgery, but he had never hoped any operation. His poor nutritional status gradually further declined, and he died due to massive hemorrhage from the tumor. In the autopsy, the histological findings revealed that the tumor was predominantly composed of high-grade spindle cell sarcoma and contained a small amount of epithelial component which was a granular cell variant of ameloblastoma. Metastatic tumors were seen in the bilateral lungs and the liver. They contained only the sarcomatous element.
Ameloblastic fibrodentinosarcoma and ameloblastic fibro-odontosarcoma: A systematic review
2018, Journal of Stomatology, Oral and Maxillofacial Surgery
Citation Excerpt :
And Gatz et al. [16] reported a case of AFOS in patient that presented a hamartomatous complex odontoma, not an AFO, 6 years earlier. Some cases of AFDS and AFOS showed large areas of the resected tumor with microscopic features of a benign tumor [17,19]. It is therefore important that a histological evaluation of the whole specimen should be carried out, because in AFDS and AFOS the epithelial component is very similar to the one in AFD and AFO, respectively, but the mesenchymal component may show increased cellularity with a sarcomatoid nature in isolated areas.
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 in a pediatric patient
2016, European Annals of Otorhinolaryngology, Head and Neck Diseases
Citation Excerpt :
AFOS is extremely rare. To our knowledge, only 14 cases, including the present case, have been reported in the literature [4–8]. Five occurred in men, 8 in women, and in one case gender was not specified.
Ameloblastic fibro-odontosarcoma is an extremely rare subtype of odontogenic sarcoma, with only 13 cases reported in the literature.
A 4-year-old male presented with a painless mandibular swelling, which appeared 4 months 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.
Although 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.
Ameloblastic fibrosarcoma developing 8 years after resection of ameloblastic fibro-dentinoma: A unique presentation
2015, Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology
Ameloblastic fibrodentinoma (AFD) is a rare mixed odontogenic tumor, with biological behavior similar to ameloblastic fibroma (AF). These tumors are considered less invasive than solid ameloblastomas and have low tendency for recurrence. Sarcomatous transformation of AF is rare but well-recognized. Though of the same group, ameloblastic fibrosarcoma (AFS) developing after resection of AFD has not been reported in literature. We present a case of a well-encapsulated soft tissue AFS developing 8 years after aggressive treatment of previous recurrent AFD. The unusual biological behavior of AFD and the importance of long term follow-up of such patients are emphasized.
Ameloblastic fibroma associated with a compound odontoma. Importance of the histopathological study
2013, Revista Espanola de Cirugia Oral y Maxilofacial
El fibroma ameloblástico es un tumor odontogénico mixto benigno de rara aparición, que constituye el 2% de todos los tumores odontogénicos, es de crecimiento lento, más común en niños y adultos jóvenes, compuesto por tejido conjuntivo fibroso embrionario y epitelio odontogénico primitivo, se caracteriza por la proliferación de tejido epitelial y mesenquimático. Aparece con más frecuencia en la mandíbula en zona de molares y premolares de pacientes jóvenes sin predilección de sexo, asociándose a veces a un diente incluido.
El presente artículo tiene como objetivo describir un caso clínico de un paciente en la segunda década de vida, con aparente anodoncia en el maxilar superior, que se encontraba asintomático y en el cual fue diagnosticado fibroma ameloblástico en maxilar superior, zona de incisivos anteriores, lado izquierdo, se realiza una breve revisión de la literatura y diagnósticos diferenciales, se analizan sus características clínicas e histológicas y la actitud terapéutica a tomar. El tratamiento quirúrgico conservador con extirpación seguida de curetaje parece ser la opción terapéutica más adecuada, y teniendo presente que el porcentaje de recidiva es del 18,3% principalmente debido a escisión incompleta de la lesión, se deben realizar controles radiográficos postoperatorios 6 meses después y cada año por los siguientes 5 años.
The ameloblastic fibroma is a benign mixed, rare odontogenic tumour, which accounts for 2% of all odontogenic tumours. It is slow growing, and more common in children and young adults. It is composed of embryonic fibrous connective tissue and early odontogenic epithelium, and characterised by the proliferation of epithelial and mesenchymal tissue. It appears most frequently in the jaw area of molars and premolars of young patients with no sex predilection, and is sometimes associated with an impacted tooth.
This article aims to describe a clinical case of a child in the second decade of life, with apparent anodontia in the maxilla, which was asymptomatic and later diagnosed as an ameloblastic fibroma in the left side maxilla incisors area. A brief review of the literature and differential diagnoses was carried out, including an analysis of its clinical and histological features, and the therapeutic approach to take.
Conservative surgical excision followed by curettage seems to be the most appropriate treatment option. It should be noted that the recurrence rate is 18.3%, mainly due to incomplete excision of the lesion. Radiographic controls should be performed six months postoperatively, and every year for the following five years.
Pediatric ameloblastic fibro-odontosarcoma of the mandible: A challenge of diagnosis and treatment
2013, Journal of Oral and Maxillofacial Surgery
Citation Excerpt :
It has been observed that approximately one third of AFS cases developed in a recurrent AF. Likewise, AFO is thought to be the precursor for AFOS.1,2 The present case is thought to develop from an AFO; although large areas of the resected tumor retained the microscopic features of a benign tumor, with the induction of normal-looking dentinoid and enameloid, in other areas the sarcomatoid component was dominant, with the induction of dysplastic dentinoid, cementoid, and enameloid.

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Case ReportHead and Neck OncologyAmeloblastic fibro-odontosarcoma: a case report

Abstract

Section snippets

Case report

Discussion

Int J Oral Maxillofac Surg

Oral Oncology

J Cranio-Maxillo-Facial Surg

Oral Oncology

Ameloblastic fibrosarcoma of the mandible: report of two cases and review of the literature

J Oral Path Med

Case Report
Head and Neck Oncology
Ameloblastic fibro-odontosarcoma: a case report