AMYLOID VARIANT OF CENTRAL ODONTOGENIC FIBROMA IN THE MAXILLA: A CASE REPORT

Abstract

A 52-year-old female patient presented with an asymptomatic intraosseous lesion in the anterior maxilla with 18 months of evolution. Clinically, the lesion presented a palatal dimpling. Radiographically, it was radiolucent and reabsorbed the roots of the teeth 21 and 22. Incisional biopsy was performed, and the histopathologic exam revealed islands and cords of odontogenic epithelium scattered through a fibrous stroma. Congo red staining revealed amyloid deposits and green birefringence was demonstrated with polarized light. No calcification was found. Immunohistochemical studies revealed positivity for AE1/AE3, CK-14, CK-19, and p63. CD1a was positive in scattered Langerhans cells into the epithelial elements. Differential diagnoses include amyloid variant of central odontogenic fibroma and noncalcifying Langerhans cell–rich variant of calcifying epithelial odontogenic tumor. We believe that both diagnoses describe the same entity and that our case shares features more in common with a description of amyloid/CD1a-associated odontogenic fibroma. The patient is undergoing medical treatment and remains in follow-up. A 52-year-old female patient presented with an asymptomatic intraosseous lesion in the anterior maxilla with 18 months of evolution. Clinically, the lesion presented a palatal dimpling. Radiographically, it was radiolucent and reabsorbed the roots of the teeth 21 and 22. Incisional biopsy was performed, and the histopathologic exam revealed islands and cords of odontogenic epithelium scattered through a fibrous stroma. Congo red staining revealed amyloid deposits and green birefringence was demonstrated with polarized light. No calcification was found. Immunohistochemical studies revealed positivity for AE1/AE3, CK-14, CK-19, and p63. CD1a was positive in scattered Langerhans cells into the epithelial elements. Differential diagnoses include amyloid variant of central odontogenic fibroma and noncalcifying Langerhans cell–rich variant of calcifying epithelial odontogenic tumor. We believe that both diagnoses describe the same entity and that our case shares features more in common with a description of amyloid/CD1a-associated odontogenic fibroma. The patient is undergoing medical treatment and remains in follow-up.