Challenging differential diagnosis, Rare disease, Educational Purpose only if useful for a systematic review or synthesis Dakshika Abeydeera Gunaratne, Hedley G. Coleman, Lydia Lim, Gary J. 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.
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Specialty: Head and neck surgery Objective: Rare disease 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 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. MeSH Keywords: Ameloblastoma, Mandibular Osteotomy, Neck Dissection, Odontogenic Tumors Background Ameloblastic carcinoma is a rare, aggressive, odontogenic epithelial malignancy with significant growth and metastatic potential that demands radical surgical intervention and vigilant post-operative surveillance. In the updated World Health Organisation WHO classification of Head and Neck tumors , ameloblastic carcinoma is categorized into three main subtypes; primary type, secondary type dedifferentiated intraosseous and secondary type dedifferentiated peripheral [ 1 ].
Primary tumors arise de novo, whereas the secondary type represents malignant transformation of a pre-existing well-differentiated ameloblastoma or odontogenic cyst. We present a rare case of mandibular ameloblastic carcinoma, secondary type de-differentiated intraosseous in a previously untreated ameloblastoma.
Case Report A year-old man was referred to the senior author G. Prior to his referral, the patient had undergone extensive dental review in January for right jaw discomfort which had developed in the months following extraction of the right mandibular third molar tooth Examination at the time had revealed a cystic mass in the region of teeth 47 and 48 with associated periodontal disease but without extra-oral swelling or trismus.
Through further investigation, we identified the same lesion on an OPG taken in June Figure 1B , prior to tooth 48 extraction, but it was not investigated at that time. A diagnosis of ameloblastoma, favoring the unicystic type, was made by a private pathologist. The following month, the patient underwent the first stage of conservative surgical treatment which included teeth 46 and 47 extraction and cyst decompression.
A repeat biopsy was performed at this time. Histologic examination of this tissue revealed an ameloblastic carcinoma. Further radical surgery was proposed, prior to which the patient underwent staging imaging that did not demonstrate evidence of regional or distant metastases.
Corresponding author. Received Mar 5; Accepted Dec Abstract 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.