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Amyloid Variant of Central Odontogenic Fibroma in the Mandible: A Case Report and Literature Review

Rare disease

Wataru Kakuguchi, Yoshiyuki Nakamichi, Tetsuya Kitamura

Japan Department of Oral and Maxillofacial Surgery, Hokkaido Chuo Rosai Hospital, Iwamizawa, Hokkaido, Japan

Am J Case Rep 2020; 21:e925165

DOI: 10.12659/AJCR.925165

Available online: 2020-07-22

Published: 2020-08-30


#925165

BACKGROUND: Odontogenic fibroma is a rare mesenchymal odontogenic tumor. It can be classified as central odontogenic fibroma (COF) or peripheral odontogenic fibroma (POF) based on clinical features. There are several variants of COF, including amyloid, ossifying, and giant cell. It grows slowly and exhibits painless cortical expansion of the jawbone. Radiographically, COF appears as a radiolucent unilocular or multilocular lesion with well-defined borders. COF may be associated with unerupted or displaced teeth and root resorption.
CASE REPORT: A 35-year-old man was referred to our hospital for submandibular swelling. Panoramic radiography and contrast-enhanced computed tomography revealed a unilocular area of bone resorption with a well-defined border and equal enhancement from the canine to first molar on the right side of the mandible. Root resorption of the first premolar and root separation of the first and second premolars were also evident. The lesion was asymptomatic. Histopathological examination of a biopsy of the lesion was suggestive of OF. Enucleation of the tumor, curettage of the bone around the tumor, and extraction of the premolar were then performed. Histopathological examination of the tumor showed fibrous connective tissue with inactive-looking odontogenic epithelial islands and strands, amyloid deposit, intercalation of Langerhans cells into the tumor epithelium, and no calcification. The final diagnosis of amyloid variant of COF was made. The postoperative course is going well.
CONCLUSIONS: Herein we describe and discuss the clinical, radiological, and pathological features of the amyloid variant of COF. This report will enhance understanding of this extremely rare variant.

Keywords: Amyloid, Fibroma, Odontogenic Tumors, Mandibular Diseases, Langerhans cells



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