Please cite this article in press as: Nagori SA, et al. Ameloblastic fibrosarcoma developing 8 years after resection of ameloblastic fibro- dentinoma: A unique presentation. J Oral Maxillofac Surg Med Pathol (2014), http://dx.doi.org/10.1016/j.ajoms.2013.11.009 ARTICLE IN PRESS G Model JOMSMP-247; No. of Pages 4 Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2014) xxx–xxx Contents lists available at ScienceDirect Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology j o ur nal ho me pa ge: www.elsevier.com/locate/jomsmp Case Report Ameloblastic fibrosarcoma developing 8 years after resection of ameloblastic fibro-dentinoma: A unique presentation Shakil Ahmed Nagori a , Anson Jose a , Ongkila Bhutia a,* , Ajoy Roychoudhury a , Aanchal Kakkar b a Department of Oral and Maxillofacial Surgery, Centre for Dental Education and Research, All India Institute of Medical Sciences, New Delhi 110029, India b Department of Pathology, All India Institute of Medical Sciences, New Delhi 110029, India a r t i c l e i n f o Article history: Received 25 September 2013 Received in revised form 20 November 2013 Accepted 29 November 2013 Available online xxx Keywords: Odontogenic Sarcoma Malignancy Soft-tissue a b s t r a c t 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. © 2013 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.  1. Introduction Ameloblastic fibroma (AF), ameloblastic fibrodentinoma (AFD) and ameloblastic fibroodontoma (AFO) are a group of benign mixed odontogenic tumors defined by WHO as “neoplasms composed of proliferating odontogenic epithelium embedded in a cellular ectomesenchymal tissue that resembles dental papilla, and with varying degrees of inductive changes and dental hard tissue for- mation” [1]. Of the group, AFD is a rare tumor commonly seen in younger age group involving the posterior mandible and is usually associated with an unerupted tooth. Histologically it is similar to AF but for the presence of dentin or dentinoid tissue, whereas AFO shows presence of both enamel and dentin. Ameloblastic fibrosarcoma (AFS), ameloblastic fibrodenti- nosarcoma (AFDS) and ameloblastic fibrodontosarcoma (AFOS) are considered malignant counterparts of these tumors and demonstrate a benign epithelial and a malignant mesenchymal component [2]. To the best of our knowledge, up to 2013 only 76  AsianAOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathol- ogy; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants. * Corresponding author at: Department of Oral and Maxillofacial Surgery, Centre for Dental Education and Research, All India Institute of Medical Sciences, Ansari Nagar, New Delhi 110029, India. Tel.: +91 9313349564. E-mail address: dr ongkila@rediffmail.com (O. Bhutia). cases [3–8] of ameloblastic fibrosarcoma have been published in lit- erature. About half of these tumors arise from previous AF while the rest arise de novo [3]. Though of the same group, AFD transforming into AFS has not been reported in literature. This article describes a unique presentation of a complete soft tissue AFS developing 8 years after aggressive resection of AFD. 2. Case report A 23-year-old male reported in July 2000, for diagnosis of a 2- month old swelling in the left angle – body region of the mandible. The general physical condition was good and medical history was non-contributory. He gave no history of pain or paraesthesia. On palpation, the swelling was non-tender, bony hard expanding the buccal and lingual cortices, with no lymph node involvement. Intra- orally, there was mild non-tender swelling in the buccal vestibule in the lower left third molar region. The overlying oral mucosa was smooth and intact. There was no associated tooth mobility and no missing tooth. Panoramic radiograph demonstrated a well-defined multilocular radiolucent lesion extending from the 2nd premolar up to the horizontal ramus just involving the apices of the molar teeth with occasional minute radiopacities. Computed tomography (CT) scan showed a well circumscribed multiloculated expansile lytic lesion with only a few specs of calcification (Fig. 1). An inci- sional biopsy specimen revealed islands and strands of odontogenic epithelium with surrounding fibrous connective tissue stroma. Dentin formation was seen in multiple sections, embedding the 2212-5558/$ – see front matter © 2013 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.  http://dx.doi.org/10.1016/j.ajoms.2013.11.009