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PubMed2004

Calcifying odontogenic cyst--Gorlin's cyst--report of two cases.

The authors present two cases of calcifying odontogenic cysts, which were confirmed by histological examination. In the first case the radiographic findings and clinical status did not indicate the presence of a calcifying odontogenic cyst. In the second case, differential diagnosis included COC. The histopathological findings showed that what appeared to be simple cases of bone translucencies was in fact an unusual odontogenic lesion. The two cases point out the possibility of incorrect assessment of deceptively banal cases in daily specialist practice.

Adult↗
PubMed1997

Features of odontogenesis and expression of cytokeratins and tenascin-C in three cases of extraosseous and intraosseous calcifying odontogenic cyst.

To characterize further the nature of calcifying odontogenic cyst (COC), we studied histologically and immunohistochemically an extraosseous and two intraosseous lesions. The extraosseous COC was in continuity with the stratified squamous epithelium of the alveolar mucosa. Immunostaining with monoclonal antibodies showed reactivity of both low- and high-molecular-weight cytokeratins, the degree of coexpression decreasing with the increasing morphological diversity of the cyst/tumour epithelium. Staining for the matrix glycoprotein tenascin-C was seen not only in the connective tissue, where its distribution patterns corresponded to the stage of hard tissue formation, but also in epithelial elements. The staining patterns were analogous to those described during normal tooth formation. Both the morphological characteristics and expression patterns of the various cytokeratin types and tenascin-C implied that COC represents a pathological counterpart of normal odontogenesis. In the case of the extraosseous COC, the correspondence could be traced back to early stages of tooth development.

Adolescent↗
PubMed1999

Initiation of ectopic epithelial calcification in a calcifying odontogenic cyst.

Ultrastructural observation was performed on a calcifying odontogenic cyst (COC) associated with an odontoma and arising in the right mandibular region of an 8-year-old Japanese boy. Four types of cells were identified in the epithelial layer of the COC. The basal cells were low columnar in shape and contained some intracellular organelles. They were attached to the neighboring cells with a few desmosomes and resembled inner enamel epithelium of the normal enamel organ. The stellate reticulum-like cells, polygonal in shape, possessed desmosomes and many cytoplasmic projections. Some intracellular organelles and a few bundles of tonofilaments were observed in the cytoplasm. The light oval cells that were pale staining with toluidine blue contained dilated membranous organelles and many relatively evenly distributed tonofilaments. These cells were usually scattered in the vicinity of the focal accumulations of ghost cells, and the cell membrane was discontinuous in parts. The ghost cells contained many bundles of tonofilaments that were 60-240 nm in diameter and arranged in various directions. No intact intracellular organelles were noted in the cytoplasm. They were attached to the neighboring ghost cells with some desmosomes and their cell membrane was discontinuous in parts. A variety of vesicles, 90-450 nm in diameter, were scattered among the tonofilament bundles. Some of these contained needle-like crystals that were considered to be initial calcification sites in ghost cells. These vesicles presented morphological similarities to matrix vesicles, and it is therefore suggested that matrix vesicle-like structures are deeply involved with initiation of calcification of ghost cells in COC.

Calcinosis↗
PubMed2004

Calcifying odontogenic cyst with ameloblastic fibroma: report of three cases.

Although it is a rare event, odontogenic tumors such as ameloblastoma, ameloblastic fibroma (AF), ameloblastic fibro-odontoma, and odontoma have been reported associated with calcifying odontogenic cyst (COC). There are only four cases of COC with AF cited in the English literature. However, three of these four cases were either included in a review of a series of cases or reported as an abstract, and limited clinical and histological information was provided. We present three additional cases of COC with AF and discuss the management for this combined lesion. Because COC is known for its histologic diversity and variable clinical behavior, and the clinical significance of an association of COC with AF is still unknown, we think it is valuable to report COC with AF with detailed clinical and pathological documentation.

Adolescent↗
PubMed2003

Calcifying odontogenic cyst: clinicopathological features and immunohistochemical profile of 10 cases.

BACKGROUND: Calcifying odontogenic cyst (COC) is an uncommon odontogenic lesion with few studies describing its immunohistochemical profile and proliferative activity reported in the literature. METHODS: Clinical and histological features and immunohistochemical expression of cytokeratins, Mel-CAM (CD146), bcl-2, PCNA and ki-67, in 10 cases of COC were studied. RESULTS: All 10 cases affected the maxilla, eight intraosseous and two peripheral. Five central cases were cystic and three were cystic associated with odontoma, and the two extraosseous showed solid histological pattern; immunohistochemistry was positive for cytokeratins 8, 14, 19, AE1/AE3 and 34betaE12 and bcl-2 in all cases, and Mel-CAM in six cases. Proliferative activity was greater in the epithelium of central cystic COC in relation to COC associated with odontoma and peripheral lesions. CONCLUSION: Calcifying odontogenic cysts showed odontogenic cytokeratin profile and bcl-2 and Mel-CAM expression indicate that these proteins may be involved in the development of COC. There were no recurrences after surgery, irrespective of their proliferative activity.

Antigens, CD↗
PubMed1998

So-called calcifying odontogenic cyst: review and discussion on the terminology and classification.

The so-called calcifying odontogenic cyst (COC) shows extensive diversity in its clinico-histopathological appearances and biological behaviour. Because of this diversity, there has been confusion and disagreement on the terminology and classification of this lesion. The attempts at classification of COC may be divided into two concepts. The first concept is the "monistic" one that all COCs are neoplastic in nature, even though the majority are cystic in architecture and appear to be non-neoplastic. The second is the "dualistic" concept that COC contains two entities: a cyst and a neoplasm. Although the World Health Organization (WHO) classified COC as an odontogenic tumour in 1992 based on the former concept, current thinking favors strongly the latter one. In this article, several previous classifications of COC in the literature are discussed and a new simple classification scheme based on the "dualistic" concept is proposed.

Biology↗
PubMed2003

Beta-catenin mutations are frequent in calcifying odontogenic cysts, but rare in ameloblastomas.

We have reported previously that alterations to beta-catenin occur frequently in adamantinomatous craniopharyngioma. Based on its histological resemblance to some odontogenic tumors, we suspected the presence of common genetic alterations among these tumors. To address this issue, 11 cases of calcifying odontogenic cyst (COC) and 20 cases of ameloblastoma were investigated for the presence of beta-catenin mutations and beta-catenin expression. Ten COCs were successfully analyzed by direct sequencing, and nine of them were found to harbor somatic beta-catenin mutations. Immunohistochemically, all of the COCs showed nuclear and cytoplasmic expression of beta-catenin with a heterogeneous pattern. No beta-catenin mutations were found in ameloblastomas, except for one case of the follicular type. All follicular ameloblastomas exhibited moderate nuclear and cytoplasmic accumulation of beta-catenin, in contrast to the predominantly membranous expression seen in the plexiform type. beta-Catenin mutation is considered to be a characteristic genetic feature of COC, and may play a critical role in its histogenesis. Although ameloblastoma closely resembles COC histologically, the two have genetically distinctive features.

Adolescent↗
PubMed1984

Recurrent calcifying odontogenic cyst.

This article reports a recurrence of a calcifying odontogenic cyst which occurred 5 years after the initial treatment. To date all recurrences of calcifying odontogenic cysts have been in elderly persons. The present recurrence was in an 11-year-old girl.

Child↗
PubMed1995

Central calcifying odontogenic cyst. Case report.

A case of central (intraosseous) calcifying odontogenic cyst affecting the mandible is reported. The clinical, radiographic, and histological features are reviewed and discussed.

Diagnosis, Differential↗
PubMed1990

Odontogenic ghost cell tumours. The neoplastic form of calcifying odontogenic cyst.

The tumourous form of the calcifying odontogenic cyst is a rare entity and only a small number cases have been described in the medical literature published in English. Our report concerns a 66-year-old patient with a tumourous mass in the maxilla, with clear COC histological features, in addition to an infiltrative growth pattern of the epithelial islands and ghost cells invading the subjacent stroma, oral mucosa and peripheral maxillary bone. Cytoplasmic and nuclear pleomorphism and atypical mitoses were rarely found. An aggressive surgical approach produced a satisfactory result. We have reviewed the literature available in English and compared it with our case.

Aged↗
PubMed1975

Calcifying odontogenic cyst. A review and analysis of seventy cases.

Six new cases of the calcifying odontogenic cyst are presented. These and sixty-four previously reported cases have been studied to further delineate characteristic clinical, radiographic, and histopathologic features of the calcifying odontogenic cyst. A review of the literature with emphasis on the possible histogenesis of the calcifying odontogenic cyst and the nature of the "ghost cells" and their products is also presented.

Adolescent↗
PubMed1998

Computed tomographic features of calcifying odontogenic cysts.

OBJECTIVES: To describe the CT appearances of 4 cases of the calcifying odontogenic cyst (COC) with particular reference to the effect of varying the window settings. METHODS: Conventional radiographs and CT scans of 4 calcifying odontogenic cysts were analyzed with respect to the presence of an impacted tooth, root resorption and calcification. In addition, increased attenuation by desquamated keratin was examined by varying the window settings on CT. RESULTS: All lesions were located in the maxilla and on conventional radiographs, had unilocular radiolucency with a well-defined margin. Calcifications and inclusion of an impacted tooth were seen in all cases. Root resorption was observed in two cases, but was not prominent. On CT, calcification was detected at the periphery of the lesion and/or around the impacted tooth in all cases. Varying the window setting revealed an increased attenuation area due to desquamated keratin. CONCLUSIONS: Varying the window setting on CT is useful as a means of identifying both desquamated keratin and peripheral calcification in COC.

Adolescent↗
PubMed2002

[The nature of calcifying odontogenic cysts of the jaws].

OBJECTIVE: To clarify the clinicopathological and behavioral spectrum of the so-called calcifying odontogenic cysts (COC). METHODS: Retrospective analysis of 21 cases previously diagnosed as COC was undertaken to evaluate their clinical, radiographic, pathological and behavioral features. RESULTS: The lesions of this series were divided into three groups, including simple cysts, benign and malignant tumors. The cyst group was composed of 16 cases (9 men, 7 women). The age of the patients peaked at the second decade, with a predilection for the premolar region of the jaws. The clinicopathological features of this group were consistent with that of developmental odontogenic cysts. Follow-up of the 13 patients treated by enucleation revealed no recurrence. The benign tumor group consisted of 4 cases with variable clinicopathologic features. Two cases were solid tumors containing ameloblastomatous epithelium, ghost cells and calcification foci. The other two cases had lesions that contained typical areas of COC and other types of odontogenic tumors (1 ameloblastoma and 1 odontogenic fibromyxoma). All the 4 cases occurred in the mandible with a relative large size. Two of which had a history of multiple recurrences. Only one case was identified as malignant tumor based on its infiltrative growth pattern and histological features. CONCLUSIONS: The so-called COC, previously recognized as a pathologic entity, can show extreme diversity in its clinical and histopathological features as well as in its biological behavior. Thus, the subgroups as simple cyst, benign and malignant tumors should be clarified and treated accordingly. Their terminology and classification should be reconsidered.

Adolescent↗
PubMed1998

Proliferative activity of calcifying odontogenic cysts as evaluated by proliferating cell nuclear antigen labeling index.

The calcifying odontogenic cyst (COC) presents with diverse histological features; thus, several subclassifications have been proposed. To evaluate the significance of the various histological features and subtypes of COC from the perspective of proliferative activity, the proliferating cell nuclear antigen (PCNA) labeling index (LI; the percentage of positive nuclei) was assessed immunohistochemically in 25 cases of COC (21 benign and four malignant). All of the benign cases were of the cystic variety and further subclassified into non-proliferative subtype (NPS; four cases); proliferative subtype (PS; eight cases); and COC associated with odontoma (COCaO, nine cases). The PCNA LI of the malignant COC (65.2+/-5.6) was significantly higher than that of the benign COC (11.6+/-9.0; P = 0.002). Non-proliferative subtype (6.8+/-2.8) showed the lowest PCNA LI and PS (17.2+/-11.2) the highest of among the three subtypes of benign cystic COC (P = 0.028). In nine cases of COCaO, six showed epithelial lining of the non-proliferative type as NPS and the other three had lining with proliferative features as PS. The PCNA LI of the latter COCaO group (14.3+/-6.6) was significantly higher than that of the former (6.1+/-4.3; P = 0.05), as seen between PS and NPS. These results demonstrate that PCNA LI is a possible parameter for differentiating malignant COC from benign COC and, whatever the subtypes, the proliferative features in the lining are the main factor influencing the proliferating activity of COC.

Cell Division↗
PubMedDate not supplied

Calcifying odontogenic cyst associated with complex odontoma: case report and review of the literature.

We report a calcifying odontogenic cyst associated with odontoma (COCaO) and an included permanent canine in the superior maxilla, in a 19 year-old-man. The calcifying odontogenic cyst (COC) was first described as a distinct entity by Gorlin et al in 1962. The lesion is a mixed odontogenic benign tumor, and although most of the cases present cystic characteristics, a few are of the solid type (15%), and its rare malignant transformation is well documented. The COC may occur in association with other odontogenic tumors, the most common is the odontoma, occurring in about 24% of the cases. For this association the term Odontocalcifying odontogenic cyst has been suggested. Radiographically is a well defined mixed lesion and histologically consists of a large cyst. In the central area of the cyst enamel and dentin deposits can be found, irregularly distributed in areas and in other parts it takes on a well defined organoid aspect. A thorough review of literature takes place and the pathogenesis is discussed.

Child↗