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.     

A case of an ameloblastic fibro-odontoma arising from a calcifying odontogenic cyst

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.     

The calcifying ghost cell odontogenic tumor – or the calcifying odontogenic cyst

Abstract. The paper reports on two new cases of calcifying odontogenic cysts, and gives a survey of 50 cases reported in the literature. In one case the ultrastructure of the characteristic ghost cells was examined. Most of the cells were anuclear or contained pyknotic nuclei. The only organelles found were very thick electro-dense fibrils of uniform size sharply defined against large empty spaces. Small needle-like crystalloid structures were seen in most cells. The ghost cells lying free in the connective tissue or embedded in the osteiod material had either lost their plasma membranes entirely or only remmants of it could be found. Histologically there is a marked similarity between the published cases. The epithelium forming either cysts or islands shows a distinct basal layer of columnar cells. Above the basal layer the cells are often stellate. Ghost cells are observed. Scattered both in the epithelium and in the surrounding connective tissue. Outeoid or dentinoid material was observed close to the ghost cells in 14 of 36 cases. Calcified deposits have been described in the ghost cells in 26 out of 36 cases reported. The material demonstrates an almost equal sex distribution of the tumor, and an almost equal distribution between the anterior maxilla and mandible. Two-thirds of the tumors were intra-osseous, and one-third were extra-osseous. The tumor occurs in all age groups, with an increased incidence in the age group of 10–19 years. There is no similarity between the clinical features of this lesion and those of ameloblastoma and calcifying odontogenic tumor.
The authors interpret the lesion as a tumor or hamartoma with a marked tendency for cystic degeneration. Therefore the designation calcifying ghost cell odontogenic tumor has been proposed.     

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.     

Peripheral calcifying odontogenic cyst

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.     

Combined calcifying epithelial odontogenic tumor and adenomatoid odontogenic tumor

A case of combined epithelial odontogenic tumor associated with an unerupted maxillary canine tooth is described. The relative proportion of adenomatoid odontogenic tumor tissue and calcifying epithelial odontogenic tumor areas in a given tumor in determining the behaviour and growth potential of this entity is discussed.     

Peripheral calcifying odontogenic cyst (Gorlin cyst)

A case report of a rare peripheral calcifying odontogenic cyst is presented. The clinical, radiographic and histopathologic features of the lesion are discussed, along with the etiology, rate of occurrence and diagnosis.     

Combined epithelial odontogenic tumor: adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor

1. Two cases of a combined epithelial odontogenic tumor which had areas of AOT and CEOT were presented. 2. A review of the studies on histogenesis of the AOT revealed that the tumor probably consists of preameloblasts, stratum intermedium, and stellate reticulum. 3. A review of the studies on histogenesis of the CEOT revealed that the probable origin was in cells of stratum intermedium. 4. It is suggested that the present cases support the aforementioned theories of histogenesis and represent AOT's which contain foci of CEOT. 5. The suggested treatment is simple surgical excision.     

Ameloblastoma ex calcifying odontogenic cyst (dentinogenic ghost cell tumor)

Calcifying odontogenic cyst (COC) has shown to be of extensive diversity in its clinical and histopathological features, as well as in its biological behavior. In this report, a rare case is described of ameloblastoma ex COC (dentinogenic ghost cell tumor) and the relevant literature is briefly reviewed.     

Adenomatoid odontogenic tumor associated with calcifying epithelial odontogenic tumor

A case of odontogenic tumor which contained areas diagnostic for both adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor arising in the upper left anterior region in a 17-year-old Japanese female is reported. The histopathological observation suggested that the lesion represented primarily adenomatoid odontogenic tumor in which multiple foci of calcifying epithelial odontogenic tumor had developed.     

Intramural calcifying epithelial odontogenic tumor

The calcifying epithelial odontogenic tumor (CEOT) is a rare lesion of the jaws. It accounts for about 1% of all odontogenic tumors. The CEOT occurs primarily in the molar-premolar region of the mandible, and 52% of cases are associated with an unerupted tooth. This report describes an unusual case in a 37-year-old woman. The tumor arose in the molar area of the right mandible, appeared radiographically as a radiolucent lesion, and was thought to be a dentigerous cyst in association with an impacted first molar. The lesion was enucleated. Microscopic examination showed it to be a dental sac, within which were the 3 elements of a typical CEOT: squamoid cells with eosinophilic cytoplasm, the homogeneous eosinophilic substance, and calcium salt deposits in the form of Liesegang rings.     

Multifocal calcifying epithelial odontogenic tumor

The calcifying epithelial odontogenic tumor (CEOT), or Pindborg tumor, is a rare and benign odontogenic neoplasm that affects the jaw. The most common manifestation of CEOT is a unifocal or localized lesion of the involved jaw, which may appear clinically as a hard tissue swelling and radiographically as a mixed radiolucent-radiopaque mass. In this article, we present a unique case of CEOT affecting multiple sites in the maxilla and mandible of a 51-year-old white man. Though biopsy samples from all involved sites revealed similar histopathologic features consistent with CEOT, the fact that there was a multifocal presentation is an unusual phenomenon for CEOT and has never been reported. Multifocal odontogenic lesions are not typical but have been observed in conditions associated with known genetic mutations. For example, multiple odontogenic keratocysts are the most common feature of the inherited condition known as nevoid basal cell carcinoma syndrome. This case, however, is the first one to demonstrate that there may be a multifocal variant of CEOT that has not been previously recognized.     

Radiographic characteristics of the calcifying odontogenic cyst

4 additional cases of calcifying odontogenic cysts (COC) were reported, together with the radiographic interpretation of 138 cases from the literature. From the review of the literature and of our own cases, the root resorption is one of the most common diagnostic findings as is cystic radiolucency accompanied by radiopacities. Other radiographic characteristics, such as loculation, tooth divergency and bone resorption were emphasized.     

骨外型牙源性钙化上皮瘤

骨外型牙源性钙化上皮瘤极为罕见,通过复习1966～2011年英文MEDLINE与1994～2011年中文CHKD中收录的文献,仅有37例骨外型牙源性钙化上皮瘤的个案报告。根据上述资料分析,发现该瘤具有以下临床特点,就诊年龄3～71岁,平均35.8岁;男13例,女19例,男女比例为1∶1.46;肿块直径0.5～4 cm,平均1.76 cm;好发部位主要是牙龈,累及下颌与上颌牙龈的比例为1∶1;组织学特征表现与骨内型牙源性钙化上皮瘤基本相同;复发率为5.4%。因该瘤具有浅表骨侵蚀能力,应行完整的肿瘤切除结合骨面刮治术。     

Radiographic analysis of a calcifying odontogenic cyst

The Radiographic appearance of 33 cases of calcifying odontogenic cyst (COC) from 1971 to 1998 were analyzed. COC has a variety of radiographic appearances. According to the WHO classification¹⁾, such cysts can be divided into five subtypes in order to be considered as central COC and one peripheral type. The subtypes are as follows: I: Unilocular, II: COC associated with odontoma, III: alveolar bone type, IV: compound type, V: recurrences that have become malignant. Ghost cells are an important feature in the diagnosis of COC. However, ghost cells may occur in other tumors as well. Differentiation of the types of COC according to radiographic appearance will aid the clinical diagnosis and treatment of odontogenic cysts.