A Case of an Ameloblastic Fibro-Odontoma Arising from a Calcifying Odontogenic Cyst.

Oral Health Science Center, Department of Pathology, Tokyo Dental College, Chiba, Japan.
The Bulletin of Tokyo Dental College 03/2001; 42(1):51-5. DOI: 10.2209/tdcpublication.42.51
Source: PubMed

ABSTRACT This case report describes an ameloblastic fibro-odontoma arising from a calcifying odontogenic cyst (COC) in the mandible of a twenty-three-year old male. The patient was referred to the Department of Oral Surgery, Tokyo Dental College, on March 30th, 2000, complaining of a painful swelling, which had appeared three weeks earlier on his left mandibular molar region. In a pathological view, the lesion was a round cyst the size of a chicken-egg, dark red in color, and surrounded by a thick membrane. The cyst had an epithelium of varying thickness which included many ghost cells and an enamel-like structure on the inside, and a thick wall of connective tissue with an ameloblastic fibro-odontoma on the outside. Enamel organ-like epithelial islands were structured radially in the form of strands with immature dentin. Cytokeratin 19 was strongly immunoreactive in the epithelium of the lesion; osteopontin and osteocalcin reacted in the mesenchymal cells and weakly in the epithelial element of this tumor.

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    • "They tend to form small clusters or large masses. Although characteristic of calcifying cystic odontogenic tumors (GGoT) [4], ghost cells are also found in other odontogenic lesions namely ameloblastoma [5] odontoma [6] and ameloblastic fibro-odontoma [7], and in nonodontogenic tumors such as pilomatrixoma [8], a tumor with hair matrix cell differentiation, and craniopharyngioma, a tumor of the pituitary gland [9]. Several theories of ghost cell formation have been put forth including that these cells are most likely abnormal keratinized bodies, or they might represent simple cell degeneration or a form of enamel matrix; or might be apoptotic odontogenic cells or represent different stages of normal and abnormal keratin formation resulting from metaplastic transformation of odontogenic tumors [4]. "
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    ABSTRACT: Notch signaling is an evolutionarily conserved mechanism that enables adjacent cells to adopt different fates. Ghost cells (GCs) are anucleate cells with homogeneous pale eosinophilic cytoplasm and very pale to clear central areas (previous nucleus sites). Although GCs are present in a variety of odontogenic lesions notably the calcifying cystic odontogenic tumor (CCOT), their nature and process of formation remains elusive. The aim of this study was to investigate the role of Notch signaling in the cell fate specification of GCs in CCOT. Immunohistochemical staining for four Notch receptors (Notch1, Notch2, Notch3 and Notch4) and three ligands (Jagged1, Jagged2 and Delta1) was performed on archival tissues of five CCOT cases. Level of positivity was quantified as negative (0), mild (+), moderate (2+) and strong (3+). Results revealed that GCs demonstrated overexpression for Notch1 and Jagged1 suggesting that Notch1-Jagged1 signaling might serve as the main transduction mechanism in cell fate decision for GCs in CCOT. Protein localizations were largely membranous and/or cytoplasmic. Mineralized GCs also stained positive implicating that the calcification process might be associated with upregulation of these molecules. The other Notch receptors and ligands were weak to absent in GCs and tumoral epithelium. Stromal endothelium and fibroblasts were stained variably positive.
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    ABSTRACT: The so-called calcifying odontogenic cyst (COC) represents a heterogeneous group of lesions that exhibit a variety of clinicopathologic and behavioral features. Because of this diversity, there has been confusion and disagreement on the terminology and classification of these lesions. We reviewed the clinicopathologic features of 21 intraosseous cases that were previously diagnosed as COC or under related diagnostic terms. Based on the biologic behavior, the lesions of the present series were divided into three subgroups: cyst, benign tumor, and malignant tumor. Sixteen cases (nine men and seven women) proved to be unicystic lesions with (five cases) or without associated odontoma. The lining epithelium of the cystic lesions fulfilled the histologic criteria for COC proposed by the World Health Organization, and their overall clinicopathologic features were consistent with that of developmental odontogenic cysts. The age of patients from the cyst group peaked at the second decade. The maxilla was affected more often (69%) than the mandible, with a predilection for the canine-premolar region (62.5%). Thirteen patients with follow-up information revealed no recurrence following enucleation. The four cases in the benign tumor group had variable clinicopathologic features. Two cases were solid tumors consisting of ameloblastoma-like sheets of odontogenic epithelium that contained ghost cells/calcification foci and juxtaepithelial dentinoid. Both patients experienced multiple recurrences following conservative surgeries. The other two lesions contained typical areas of COC and other types of odontogenic tumors (one ameloblastoma and one odontogenic myxofibroma). All four lesions occurred in the mandible and were relatively large. In the present series one case identified as malignant tumor arose from a previously benign COC. The tumor shared some features of COC (ghost cell foci and dystrophic calcification) but also had prominent mitotic activity, nuclear and cytoplasmic pleomorphism, areas of tumor necrosis, and infiltrative/destructive growth. Recognizing the extreme diversity in clinicopathologic features and biologic behavior among the so-called COCs, we suggest that the term COC should be used to specifically designate the unicystic lesions with or without an associated odontoma, i.e., lesions of the cyst group, and other related lesions identified as benign tumor and malignant tumor should be termed and classified separately. A tentative scheme with respect to the terminology and classification for this group of disparately behaving lesions was herein proposed to reflect the likely difference of their nature.
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