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Calcifying odontogenic cyst is rare; about 140 cases have been described in the literature. The problem of the proper classification of the calcifying odontogenic cyst has been discussed almost since its identification. Recently it has been concluded that the group of lesions designated as calcifying odontogenic cyst contained two entities, a cyst and a neoplasm.
Calcifying odontogenic cysts (COCs) represent a group of lesions that may be broadly classified into two main entities: cysts and neoplasms. In the present study 30 non-neoplastic cystic COCs were examined by a quantitative histological method in an attempt to calibrate the relative distribution of the type of epithelial lining, intensity of ghost cell formation and the amount of dentinoid present. The results showed that there are two main types of cystic COC: an odontoma-producing type and a non-odontoma-producing variant. Morphologically, tooth-like structures were a valid distinguishing feature, while morphometrically the odontoma-producing variant showed a greater amount of luminal and mural dentinoid as well as luminal ghost cells. Demographic analysis also revealed that the odontoma-producing COC occurred in younger patients and showed an even sex distribution, whereas the non-odontoma-producing type was seen in older patients and showed a predilection for females. Both subtypes were more prevalent in the Chinese population and occurred preferentially in the maxilla.
Calcifying odontogenic cyst is a rare benign lesion of the jaws, with no particular specificity for age, sex or site. Three new cases are presented together with a review of the radiological literature. There appear to be no pathognomonic radiological features, although a superficial location on the affected bone and the presence of internal calcification may be useful signs. The role of radiology appears to be limited to determining lesion extent prior to surgery.
The calcifying odontogenic cyst may present some problems of differential diagnosis, both clinically and histopathologically. The present article reports the case of a calcifying odontogenic cyst in the maxilla of a 54-year-old woman. The histopathologic examination showed that structures characteristic of calcifying odontogenic cyst and odontogenic tumor of ameloblastoma-like appearance occurred simultaneously. The lesion was benign. After surgical removal of the cyst, the wound healed uneventfully and the region was free of symptoms 12 months postoperatively.
The calcifying odontogenic cyst (COC) has been reported to be associated with odontoma in about 24% of cases. Separation of the cases of COC associated with odontoma (COCaO) may lead to a better understanding of the pathogenesis of this lesion. A screen of the literature revealed 52 cases of COCaO. The male to female ratio was 1:1.9, with a mean age of 16 years. The most common location was the maxilla (61.5%). The radiographic appearance of most cases (80.5%) was of a well-defined, mixed radiolucent-radiopaque lesion. Histologically, the lesions usually consisted of a single large cyst (which is similar to simple COC) with tooth-like structures that appear to be an integral part of the lesion, giving the impression of a single lesion. In addition to the unique histologic features, differences in gender and in distribution were found between the cases of COCaO and those of simple COC. COCaO may be regarded as a separate entity and classified as a benign, mixed odontogenic tumor. The term odontocalcifying odontogenic cyst is suggested.
BACKGROUND: Calcifying odontogenic cyst (COC) is a rare lesion representing about 1% of jaw cysts. It may occur in a central (intraosseous) or peripheral (extraosseous) location. METHOD: A case of peripheral COC located on the gingiva, appearing as a painless, circumscribed, pink nodule has been reported. RESULTS: Peripheral, in contrast to central, COC tends to affect older patients. Peripheral COC is a less aggressive lesion than the central counterpart, and a simple excision biopsy is curative. CONCLUSION: The histological finding of a keratinized epithelium rich in ghost cells has helped in making the diagnosis.
The ultrastructural features of calcification in a case of calcifying odontogenic cyst (COC) were studied. Scanning electron microscopic examination of the inner parts of the cyst wall revealed many short microvilli, and X-ray microanalysis of the high-density masses in the intercellular parts showed prominent calcium peaks, which meant that these masses were calcified materials. On transmission electron microscopic observations, many calcifications exhibited a distinctive ring formation around the periphery of a central core that consisted of an amorphous structure. These calcifications were observed with necrotic remnants of nuclear material and many identifiable mitochondria, thin fibers, and epithelial cells. The cytoplasm of ghost cells consisted of numerous short electron-dense tonofilament bundles. Needle-like structures were shown in the tonofilament bundles. X-ray diffraction analysis showed that the needle-like crystals were hydroxyapatite. It is suggested that calcification in a COC may be related to degenerative mitochondria and tonofilament bundles of ghost cells.
The calcifying odontogenic cyst (COC) or Gorlin cyst is a rare and benign lesion most often intraosseous, although an appreciable number of cases are peripheral. Two histologic entities are described, one being cystic and the other neoplastic. The cystic type may occur as three variants. The occurrence of ghost cells, although shared with calcifying epithelioma of Malherbe, craniopharyngioma and other odontogenic tumors, represents the most conspicuous feature of the COC. Ghost cells are so called because they stain only faintly with common dies, including eosin. Although seldom studied, it is claimed that ghost cells are keratinized. However, this hypothesis is not universally accepted since electron microscopic studies give evidence that ultrastructural features of ghost cells differ from what is observed during the keratinizing process of the epidermis and oral mucosae. The present study, dealing with one case of COC, combines two complementary techniques: one a Rhodamine B keratin specific staining method and the other an immunohistochemical technique based on the use of a primary antiserum directed against high molecular weight keratins. With the Rhodamine B method, ghost cells and orthokeratinized cells of control gingiva are strongly stained while intermediate cells of the COC are less prominent. On the other hand, with the immunohistochemical technique, intermediate mediate cells of the COC are less prominent. On the other hand, with the immunohistochemical technique, intermediate cells of the COC and high level cells of control epithelium react strongly. Ghost cells of the COC are only faintly labelled and orthokeratinized cells of the control gingiva remain unlabelled. In combination with previous histochemical studies, these results confirm the occurrence of a keratinizing process.(ABSTRACT TRUNCATED AT 250 WORDS)
The calcifying odontogenic cyst (COC) is a rare lesion without specific clinical characteristics. Its diagnosis is essentially histopathological showing the presence of ghost cells associated with narrow squamous epithelium, the basal strata of which consist of clearly delineated cells differentiating in areas of the stellate reticulum in a similar way to ameloblastoma. The results of ultrastructural observations of the ghost cells as well as histochemical and immunohistochemical studies suggest that they are the sites of abnormal keratinisation. The aim of this study of one COC mas to locate the stages in epithelial maturation associated with the formation of ghost cells. Six monoclonal antibodies were used; three with a wide spectrum (KL1, AE3, AE1), two with a narrow spectrum against high molecular weight cytokeratins (AE2, AE8) and one against vimentin (M725). Histopathological examination of the COC revealed three different types of cells in the epithelial lining and the epithelial islands; small basal cubic cells surrounding larger ones placed centrally or suprabasally; balloon shaped cells or flattened ghost cells rolled up on themselves to resemble keratinising pearls, or cornifying cells. The ghost cells and cornifying cells had an altered distribution of their cytokeratins demonstrated by the absence of staining of antibodies against cytokeratins. The differentiated cells adjacent to them showed cytokeratins typical of squamous epithelium rather than those associated with the process of keratinization. The coexistence of cornifying and ghost cells testifies to the great potential of odontogenic epithelium to form numerous epithelial islands.
BACKGROUND: Adenomatoid odontogenic tumors (AOT) and calcifying odontogenic cysts (COC, Gorlin's cyst) are rare benign specificities of odontogenic tumors. Most odontogenic tumors are lesions of embryonic odontogenic tissue. Radiographically, odontogenic tumors may mimic an odontogenic follicular cyst. CASE REPORT: This paper describes the case of an 11-year-old female patient with a remarkable combination of these two entities. The lesions were found at the site of a retained first premolar and a persistent deciduous tooth 84 of the right lower jaw. Without clinical symptoms the tumor had expanded the vestibular cortical bone of the premolar region. Radiographically, the lesion appeared as a typical follicular cyst. During the operation, excochleation of the cyst was performed and the premolar was extracted. Finally, the epithelium of the cyst was treated by curettage. The histologic specimen revealed the diagnosis of an adenomatoid odontogenic tumor (AOT) in a calcifying odontogenic cyst (COC). DISCUSSION: The described case shows that symptoms of odontogenic tumors are usually nonspecific. For that reason one should always take a biopsy for histological investigation when patients present atypical cysts of the jaw. In rare cases even malignant odontogenic neoplasms could occur.
A rare case of the malignant form of Gorlin's cyst is reported in a 42-year-old woman. The neoplasm developed in the right maxilla, infiltrated the anterior wall and roof of the sinus and grew into the nasal cavity. Based upon literature data we characterized various clinical and histological forms of Gorlin's cyst (calcifying odontogenic cyst).
Odontogenic tumors composed of 2 distinct types of lesions are unusual. We report an odontogenic tumor that was composed of calcifying odontogenic cyst and ameloblastic fibroma that occurred in the right posterior maxilla of a 22-year-old Korean woman. The tumor had a cystic component with an ameloblastic epithelial lining and conglomerates of so-called ghost cells, and there were deposits of dentinoid material adjacent to the cyst. These are features characteristic of calcifying odontogenic cyst. Enamel organ-like epithelial islands were observed within a dental papilla-like stroma of the cyst wall. Additionally, a solid portion of the tumor had characteristic features of ameloblastic fibroma, i.e., a myxoid cellular stroma with numerous elongated islands of ameloblastic epithelium. Ghost cell masses were found in the area of ameloblastic fibroma as well. The distribution of the ghost cells suggests that this is a hybrid lesion rather than a collision tumor.
18 Patients with calcifying odontogenic cyst underwent surgery in our Department from 1977 to 1993 were reported.8 male and 10 female aged from 11 to 52 with medium or 33 years were included.16 cases were pathologically confirmed as primary intraosseous cyst(central),and 2 as extraosseous cyst(peripheral).There was no recurrence after the local excision for preripheral calcifying odontogenic cyst and local curettage for central one.There were 3 in 6 cases of tumor typed cyst who had recurred postoperatively,two of them had recurred repeatedly and cured by means of local radiotherapy.The authors recommend that the tumor type of intraosseous calcifying odontogenic cyst be classified as a border-line tumor and treated as well as the principles of low malignant tumor.Post-operative radiotherapy could be used for recurrent cases.
The central calcifying odontogenic cyst (CCOC) is an uncommon lesion. The present study reports 17 new cases of CCOC and analyses their clinical and histomorphologic features. The data revealed that CCOC were usually diagnosed in the second decade of life with an almost equal distribution between the maxilla and the mandible. Six of the 17 cases (35%) were associated with odontomas and 6 lesions (35%) were associated with unerupted teeth. Histologically, most of the lesions were unilocular cysts but at least one was multilocular and one was a mixed lesion - partially cystic and partially solid. Excision with long-term follow-up (up to 10 years) is the treatment of choice.
Calcifying odontogenic cysts (COC) were immunohistochemically described using different keratin proteins and involucrin as well as histopathology. The cystic lining epithelium was composed of calcifying, keratinizing, squamous, and columnar epithelial cells, and included calcified masses of irregular shape and various size as well as ghost cells. Calcifying epithelium gave negative or only trace staining for keratins detected with low molecular keratin (PKK1), but were regularly positive with high molecular keratin (KL1) and polyclonal antibody for keratin (TK). They were occasionally positive for involucrin. The cells located in the periphery of the calcified masses had a particular abundance of high molecular weight and total keratins (KL1 and TK). Calcified bodies and ghost cells were devoid of any immunoreactivity. Squamous epithelium was relatively similar to that of normal squamous cell epithelium in the oral mucosa. It were most commonly found in columnar cystic epithelial cells which displayed intense staining with all immunoreagents. It is postulated that such epithelial cells may have a strong potentiality to transform into ghost cells or to undergo metaplasia. They may develop altered synthesis of homogenous acellular materials and finally become transformed into calcifying epithelium containing dystrophic calcified masses.
Calcifying odontogenic cyst (COC) is an uncommon developmental odontogenic cyst first described by Gorlin in 1962. It is considered as extremely rare and accounts for only 1% of jaw cysts reported. Because of its diverse histopathology, there has always been confusion about its nature as a cyst, neoplasm or hamartoma. Several subclassifications have been proposed. Here, we present a case of calcifying odontogenic cyst with ameloblastic proliferation - an extremely rare histologic variant. The classical histologic features of the lining epithelium in the form of cords and presence of characteristic ghost cells were seen along with ameloblastomatous proliferations. Ameloblastomatous COC microscopically resembles unicystic ameloblastoma except for the ghost cells and calcifications within the proliferative epithelium. The nature of the COC is controversial. The case is presented here for its rarity, and difference between ameloblastomatous COC and ameloblastoma ex COC has been emphasized.
METHOD AND RESULTS: Calcifying odontogenic cysts (COCs) were examined histopathologically and immunohistochemically to characterize the histological and cytological properties of these lesions. Histopathologically, COCs showed thin or thick lining epithelium with ghost cells. COCs were classified according to proliferative type or nonproliferative type lining epithelium, the presence or absence of ameloblastomatous appearance, and the presence or absence of odontoma in the cyst walls. Immunohistochemically, amelogenin protein was expressed chiefly in ghost cells, whereas cytokeratin 19 (CK19) and bcl-2 proteins were expressed chiefly in lining epithelial cells. The proportion of cases positive for bcl-2 protein was slightly higher in COCs with odontoma than in those without odontoma. Lining epithelial cells sporadically showed positive reactions for Ki-67 antigen. Mean Ki-67 labeling index was slightly greater in COCs with proliferative type lining epithelium, COCs with ameloblastomatous appearance of the cyst walls, and COCs with odontoma of the cyst walls than in COCs without these histological features. Our results suggest that ghost cells or lining epithelial cells show ameloblastic cytodifferentiation or odontogenic epithelial characteristics, that bcl-2 protein is associated with survival of lining epithelial cells in COCs, and that high proliferation potential is associated with ameloblastomatous proliferation or combined odontoma. COCs exhibited various histological features with several transitional forms, and immunohistochemical examinations revealed little or no difference in cytodifferentiation and cellular activity among COCs. CONCLUSION: We conclude that COCs with various histological features have neoplastic potential and may not be separate entities within the same histological spectrum.
Calcifying odontogenic cyst(COC) is an uncommon developmental odontogenic cyst and was first described by Gorlin in 1962. It is considered as extremely rare and accounts for only 1% of the jaw cysts reported. Here, we present two cases of which one occurred in a 32 year old female with a swelling in the lower anterior region crossing the midline and another in a 29 year old male with a swelling in the upper anterior region without crossing the midline. The radiograph revealed a well circumscribed radiolucency in the first case and with some specks of opacities in the second case. It was not associated with any missing or impacted tooth. Histopathological examination was done with Hematoxylin and Eosin and in addition it was studied immunohistochemically for cytokeratin. The Classical histological features of lining epithelium in the form of cords, presence of ghost cells and some amount of dentinoid tissue were seen. The nature of COC is controversial. Here we have discussed the controversies regarding COC as well as the various proposed classifications for this lesion. Based on the histopathological findings, the diagnosis was confirmed as calcifying odontogenic cyst. These cases are presented here for its rarity.