Calcifying Odontogenic Cyst Research Articles

Calcifying odontogenic cyst with odontogenic keratocyst: a case report and review of the literature

The calcifying odontogenic cyst (COC), first identified as a separate and distinct lesion by Gorlin et al. in 1962, is an uncommon benign lesion, consisting of a proliferation of odontogenic epithelium and scattered nests of ghost cells and calcifications that may form the lining of a cyst or present as a solid mass. The COC occurs alone or occasionally with odontomas or other odontogenic tumors, and it is this variable histology and clinical behavior that has raised the question of whether or not it is a cyst or a true neoplasm. The odontogenic keratocyst (OKC) is a locally aggressive odontogenic cyst lined by parakeratinizing epithelium that also exhibits characteristics of a neoplasm, including rapid growth, a high rate of recurrence when treated conservatively, and the presence of a gene mutation. We describe a patient diagnosed with an OKC of the mandible that arose simultaneously with a COC of the anterior maxilla. The occurrence of 2 "cystic neoplasms" in the same patient is an occurrence which to our knowledge has not been previously reported. We discuss the significance of the case and review the current literature regarding these lesions.

Immunohistochemical analysis of p53 and PCNA expression in calcifying odontogenic cyst

Calcifying odontogenic cyst (COC) is a developmental odontogenic cyst in the jaw. Because of its diverse histopathologic features and biological behavior, there has long been confusion with regard to its nature as a cyst or neoplasm. This study evaluated the proliferative activity of 57 COC samples, including simple cyst (10 cases), cystic neoplasm (34 cases), solid neoplasm (6 cases) and combined lesion (7 cases) by p53 and PCNA immunohistochemical staining. For assessment of p53 and PCNA positivity, the number of positively stained cells with brown-stained nuclei was counted in 1000 cells from each sample. p53 and PCNA expression in the solid neoplasm subtype were significantly higher when compared to cystic neoplasm and simple cyst (P < 0.05). The lowest p53 and PCNA expression was found in the simple cyst subtype. p53 and PCNA expression in the basal and suprabasal layers was significantly higher in the solid subtype when compared to others, and the difference between COC groups was significant. The results demonstrated that within benign types of COC, the amount of p53 and PCNA in proliferative epithelium is significantly higher when compared to non-proliferative epithelium. p53 and PCNA markers are possible parameters for differentiation of COC subtypes.

Persistent apical periodontitis associated with a calcifying odontogenic cyst

To report a case of calcifying odontogenic cyst (COC) that was suggestive of apical periodontitis adjacent to the roots of the maxillary incisor teeth. Tooth 21 presented with clinical and radiographic signs of secondary infection, a post within the root canal and substantial internal tooth destruction; it was scheduled for endodontic surgery. Teeth 12 and 22 were root filled following the placement of a calcium hydroxide intracanal dressing for 21 days. Three attempts at root canal disinfection in tooth 11 were unsuccessful, and a persistent purulent drainage precluded completion of root canal treatment. Surgical enucleation of the periapical lesion was undertaken and the tissues submitted for histopathological examination. A diagnosis of COC was established based on the microscopic analysis. COC is an unusual benign lesion that represents 2% of all odontogenic lesions. Depending on the stage of development, it can mimic a large lesion associated with apical periodontitis and should therefore be considered in the differential diagnosis. In the case of COC, the definitive diagnosis can only be made with histopathological analysis. Persistent apical periodontitis may be of nonendodontic origin. * Histological examination is essential to establish the cause of persistent apical periodontitis. * Calcifying odontogenic cyst can mimic apical periodontitis.

Ghost cell odontogenic carcinoma transformed from a dentinogenic ghost cell tumor of maxilla after multiple recurrences

Dentinogenic ghost cell tumor (DGCT), also referred to as odontogenic ghost cell tumor, is a rare tumor classified as a neoplastic variant of calcifying odontogenic cyst (COC). Ghost cell odontogenic carcinoma (GCOC) is the especially rare malignant counterpart of DGCT and COC. The case of a middle-aged male with a maxillary DGCT which transformed to GCOC after 5 recurrences during a 21-year period is presented. When the diagnosis of GCOC (first identified as malignant after 17-year recurrence) was made, the tumor cells showed more obvious atypia, necrosis, and numerous mitoses, and interestingly the ghost cell was hard to find and even disappeared. The significances in pathologic diagnosis and clinical treatment are discussed.

AMELOBLASTOMATOUS CALCIFYING CYSTIC ODONTOGENIC TUMOR: A RARE HISTOLOGIC VARIANT

In agreement with the new classification of the World Health Organization (WHO) 2005, calcifying odontogenic cyst (COC) or calcifying cystic odont-ogenic tumor (CCOT) is an uncommon developmental odontogenic lesion that demonstrates histopathologic diversity. Predominantly, it occurs in the anterior region of the mouth and in the second and third decades of life. Odontogenic tumors such as ameloblastoma have been reported to be associated with CCOT. In this paper, we report a case of ameloblastomatous CCOT in a boy with involvement of mandibular ramus- an extremely rare histologic variant. The microscopic examination revealed a CCOT; ghost cell within ameloblastic islands in the connective tissue wall was observed.

Developmental odontogenic cysts of jaws: a clinical study of 245 cases.

Background and aims The aim of this study was to investigate the relative frequency of developmental odontogenic cysts in an Iranian population. Materials and methods In this study 245 cysts from both jaws, treated in the Faculty of Dentistry at Tabriz University of Medical Sciences during a 10-year period from 1998 to 2008, were analyzed in order to evaluate the incidence of such cysts. We had permission from all the patients. Case histories of 65% of male and 35% of female patients were analyzed. The age of the patients varied from 14 to 64 years, with an average of 33.21 ± 10.89. Results In this 10-year study of odontogenic cysts, 97 cases were developmental odontogenic cysts with the following inci-dence: dentigerous cyst, 44%; odontogenic keratocyst, 36%; primordial cyst, 9%; Gorlin cyst, 2%; lateral periodontal cyst, 3%; eruption cyst, 3%; and gingival cyst, 3% (adults 2%, infants 1%). A total of 60% of the cysts were found in the mandible and 40% in the maxilla. Regarding the mandible, the molar region was involved in 47% of the cases, premolar region in 33% and anterior region in 20% (total = 100%). Regarding the maxilla, the canine-to-canine region was involved in 52% of the cases, premolar region in 20% and molar region in 28% (total = 100%). Conclusion An important finding in this study was the fact that 39% of the jaw cysts were developmental odontogenic cysts and the most common developmental odontogenic cysts were dentigerous cyst and OKC (odontogenic keratocyst).

The expression of NF-κB, Ki-67 and MMP-9 in CCOT, DGCT and GCOC

Calcifying odontogenic cysts (COCs) represent a group of rare odontogenic lesions with a diversity of clinicopathological and behavioral features. According to the WHO classification of head and neck tumors in 2005, COC has been divided into calcifying cystic odontogenic tumor (CCOT), dentinogenic ghost cell tumor (DGCT) and ghost cell odontogenic carcinoma (GCOC). With few reports available on its immunohistochemical profile, this study investigated the histopathological features and the expression of nuclear factor kappaB (NF-kappaB), Ki-67 and matrix metalloproteinase-9 (MMP-9) in CCOT, DGCT and GCOC. According to the WHO classification of head and neck tumors in 2005, 26 cases of the so-called COC were diagnosed as CCOT (n=14), DGCT (n=7) and GCOC (n=5), respectively. The specimens of 26 COCs and 10 classic ameloblastomas (as control group) were examined by immunohistochemistry using anti-NF-kappaB p65, anti-Ki-67 and anti-MMP-9 antibodies and by in situ hybridization(ISH)using anti-MMP-9 mRNA probes. Immunohistochemical reactivity for NF-kappaB was mainly detected in the cytoplasm of tumor cells, and nuclear reactivity was only seen in few tumor cells in COC and classic ameloblastomas. Rate of nuclear staining was less than 1%. The expression of Ki-67 in GCOC was significantly higher than those in CCOT (p<0.001), DGCT and ameloblastoma (p<0.005). In COCs and ameloblastomas, expression of MMP-9 mRNA and protein was detected in tumor cells as well as in stromal cells. The positive staining for MMP-9 protein was detected in stromal cells of all GCOC cases and was significantly stronger than those in CCOT and DGCT groups (p<0.05). NF-kappaB may minimally affect the progression and local invasiveness of CCOT, DGCT and GCOC. GCOC show significantly higher proliferative activity than CCOT and DGCT. MMP-9 in stroma is associated with invasive ability of the CCOT, DGCT and GCOC.

Calcifying Odontogenic Cyst Associated with an Orthokeratinized Odontogenic Cyst

Odontogenic tumors composed of two or more distinct types of lesions are unusual. In this paper, a case of an odontogenic lesion characterized by simultaneous occurrence of areas of calcifying odontogenic cyst (COC) and orthokeratinized odontogenic cyst (OOC) is described. The lesion was asymptomatic and presented at the radiographic examination as a unilocular well-delimited radiolucency extending from left incisor to right premolar area in the mandible. To date, this is the first report of COC associated with an OOC.

β‐catenin gene alterations in a variety of so‐called calcifying odontogenic cysts

Calcifying odontogenic cysts (COCs) evidence a variety of histopathological features, in addition to biological behaviors. Several systems have been proposed for the classification of COCs. However, the molecular changes underlying their development remain largely unknown. In an effort to elucidate the participation of beta-catenin gene mutation in COC development, we evaluated the genetic alterations and expression of beta-catenin in different COC subtypes with archival paraffin blocks. beta-catenin gene mutations were detected in all subtypes. Immunohistochemically, all cases of COCs evidenced cytoplasmic and nuclear beta-catenin expression. Beta-catenin gene mutations and beta-catenin overexpression have been identified as characteristics of COCs. Therefore, the constitutive activation of Tcf/Lef-dependent transcription appears to be intimately involved in their development. These findings indicate that aberrations of the Wnt signaling pathway may perform a crucial role in the development and differentiation status of the odontogenic epithelium in COCs via the deregulation of cell proliferation.

Dentinogenic ghost cell tumor: A variant of Gorlin′s cyst

Calcifying odontogenic cyst (COC) was described as a distinct entity for the first time by Gorlin and his associates in 1962. Dentinogenic ghost cell tumor (DGCT) was described by Praetorius et al. in 1981 as a neoplastic variety of COC. DGCT is an extremely rare odontogenic tumor and accounts for only 2% to 14% of all COCs. A case of DGCT in a 40-year-old male patient is being reported.

Pilomatricoma: A dermal analog of calcifying odontogenic cyst

Pilomatricoma, also known as "Calcifying Epithelioma of Malherbe" is an uncommon, benign, cutaneous tumor originating from the hair matrix. Most often, its precise preoperative diagnosis is not possible. Rarity of this lesion and its histological resemblance to the calcifying odontogenic cyst (the Gorlin's cyst) make it relevant, both to the maxillofacial surgeons and the pathologists. Here, we describe a case of pilomatricoma of the periorbital region in a young Indian female patient. We believe that its inclusion is essential in the differential diagnosis of hard masses of the maxillofacial and paraoral regions.

Conservative Treatment of Calcifying Odontogenic Cyst: Report of 3 Cases

The calcifying odontogenic cyst (COC) was first defined as a separate entity by Gorlin et al in 1962. 1 Gorlin R.J. Pindborg J.J. Clausen F.P. et al. The calcifying odontogenic cyst—A possible analogue of the cutaneous calcifying epithelioma of Malherbe An analysis of fifteen cases. Oral Surg. 1962; 15: 1235 Abstract Full Text PDF Scopus (269) Google Scholar The COC represents about 1% of jaw cysts, and although it may occur in soft tissue, it is most commonly found within bone. 2 Odell E.W. Morgan P.R. Biopsy Pathology of the Oral Tissues. Chapman and Hall Medical, London, UK1998 Google Scholar , 3 Shamaskin R.G. Svirsky J.A. Kaugards G.E. Intraosseous and extraosseous calcifying odontogenic cyst (Gorlin Cyst). J Oral Maxillofac Surg. 1989; 47: 562 Abstract Full Text PDF PubMed Scopus (42) Google Scholar Both the intraosseous and extraosseous forms occur with about equal frequency in the maxilla and mandible, mainly in the incisor and canine areas. 4 Altini M. Ferman A.G. The calcifying odontogenic cyst: Eight new cases and review of the literature. Oral Surg Oral Pathol Oral Radiol. 1975; 40: 751 PubMed Google Scholar , 5 Freedman P.D. Lumerman H. Gee J.K. Calcifying odontogenic cyst: A review and analysis of seventy cases. Oral Surg Oral Med Oral Pathol. 1975; 40: 93 Abstract Full Text PDF PubMed Scopus (110) Google Scholar , 6 Nagao T. Nakajima T. Fukushima M. et al. Calcifying odontogenic cyst: A survey of 23 cases in the Japanese literature. J Maxillofac Surg. 1983; 11: 174 Abstract Full Text PDF PubMed Scopus (36) Google Scholar , 7 Buchner A. The central (intraosseous) calcifying odontogenic cyst: An analysis of 215 cases. J Oral Maxillofac Surg. 1991; 49: 330 Abstract Full Text PDF PubMed Scopus (155) Google Scholar Although the peripheral COC affects individuals in the sixth decade of life, the central variant is observed most commonly in the second decade and does not show gender predilection. 7 Buchner A. The central (intraosseous) calcifying odontogenic cyst: An analysis of 215 cases. J Oral Maxillofac Surg. 1991; 49: 330 Abstract Full Text PDF PubMed Scopus (155) Google Scholar , 8 Buchner A. Merrell P.W. Carpenter W.M. et al. Central (intraosseous) calcifying odontogenic cyst. Int J Oral Maxillofac Surg. 1990; 19: 260 Abstract Full Text PDF PubMed Scopus (24) Google Scholar

Dentinogenic Ghost Cell Tumors

Calcifying odontogenic cyst (COC) was first described by Gorlin et al in 1962 1 Gorlin R.J. Pindborg J.J. Clausen F.P. et al. The calcifying odontogenic cyst: A possible analogue of the cutaneous calcifying epithelioma of Malherbe. Oral Surg Oral Med Oral Pathol. 1962; 15: 1235 Abstract Full Text PDF PubMed Scopus (267) Google Scholar and defined by WHO 2 Kamijo R. Miyaoka K. Tachikawa T. et al. Odontogenic ghost cell carcinoma: Report of a case. J Oral Maxillofac Surg. 1999; 57: 1266 Abstract Full Text PDF PubMed Scopus (14) Google Scholar in 1971 as “a cystic lesion in which the epithelial lining shows a well-defined basal layer of columnar cells, an overlying layer that is often many cells thick and that may resemble stellate reticulum, and masses of ghost epithelial cells that may be in the epithelial cyst lining or in the fibrous capsule.” Calcifying odontogenic cyst has the features of a cyst primarily, but it also has some characteristics of a solid neoplasm. The solid variant of COC was called calcifying ghost cell odontogenic tumor formerly. The nomenclature of this tumor was changed to dentinogenic ghost cell tumor (DGCT) at 2005 World Health Organization (WHO) histologic classification of odontogenic tumors. 3 Barnes L. Eveson J. Reichart P. et al. WHO classification of tumors: Pathology and genetics of tumors of the head and neck. Oxford University Press, Lyon, France2005 Google Scholar On rare occasions, the lesion may even transform into carcinoma. 4 Lin C.C. Chen C.H. Lin L.M. et al. Calcifying odontogenic cyst with ameloblastic fibroma: Report of three cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2004; 98: 451 Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar This report describes a case of DGCT that occurred in the maxilla of a 20-year-old female.

Ameloblastomatous Gorlin's cyst

The calcifying odontogenic cyst (COC), first described by Gorlin et al. in 1962, represents a heterogeneous group of lesions that exhibit a variety of clinicopathologic and behavioral features. COC has been categorized under two basic groups namely, cystic and neoplastic. Even after several classifications and sub-classifications, COC remains an enigma. Very few cases of ameloblastomatous COC have been reported in the literature. In this report, we present a case of ameloblastomatous COC / Gorlin's cyst, emphasizing on the rarity of the lesion and distinguishing it from ameloblastomatous ex COC pictographically.

Odontogenic Ghost Cell Carcinoma of the Maxilla

This is from a 39-year-old male with a 26-six-year history of a maxillary mass, recurring after excision, 10cm in widest diameter, solid and cystic.&#x0D; &#x0D; Odontogenic ghost cell carcinomas (OGCC) are rare and only a dozen or so have been reported in the literature. This tumor appears to be more common in Asians based on the cases that have been reported and is more common in the maxilla than the mandible. Histologically, elements of a benign calcifying odontogenic cyst (COC) can be identified in all the malignant variants, either separated or admixed with the malignant epithelial component. The biological behavior of the tumour is unpredictable, with some cases characterized by relatively indolent growth and others by a locally aggressive and potentially fatal course. The tumour apparently arises most often from malignant transformation of a preexisting benign COC, although it may also develop from other odontogenic tumours. The section shows cords and islands of stellate reticulum displaying peripheral palisading of ameloblasts. There are numerous polygonal cells that have eosinophilic cytoplasm and indistinct cellular detail (“ghost cells”). Calcified dental matrix is also present. Although cytologic atypia appears mild, the borders are infiltrative.

A case report of a solid keratocystic odontogenic tumor: is it the missing link?

Our understanding of odontogenic ghost cell lesions has progressed from the original definition of a simple cyst, through the recognition of a solid variant, to our present definition of 2 neoplastic lesions, one cystic (calcifying cystic odontogenic tumor/calcifying odontogenic cyst), and the other solid (dentinogenic/odontogenic ghost cell tumor) and considered to be more aggressive. The metamorphosis of our understanding of the odontogenic keratocyst into a recognized cystic neoplasm, the keratocystic odontogenic tumor, has occurred because of its observed biologic behavior and modern investigations that have revealed chromosomal and genetic abnormalities consistent with neoplastic progression. Missing from the analogous progression is the existence of a solid variant of the keratocyst. This report documents a solid keratocystic odontogenic tumor found in the left mandible of a 52-year-old caucasian male. Based on 15 cases, Gorlin et al. in 1962 reported the calcifying odontogenic cyst (COC) as a ghost cell– containing cystic lesion of the jaws. However, it soon became apparent that ghost cell–containing odontogenic lesions were heterogeneous. In 1981, Praetorius et al. published a classification that included both cystic (type 1) and solid (type 2) lesions. Biologic behavior suggested that these lesions were neoplastic, with the latter being more aggressive and infiltrative.

Expression of nuclear factor-kappaB, Ki-67 and matrix metalloproteinase-9 in calcifying odontogenic cyst

To evaluate the expression of nuclear factor-kappaB (NF-kappaB), Ki-67 and matrix metalloproteinase-9 (MMP-9) in calcifying odontogenic cyst (COC), in order to investigate the proliferation and invasion of COC. Twenty-six cases of COC were classified into calcifying cystic odontogenic tumor (CCOT), dentinogenic ghost cell tumor (DGCT) and ghost cell odontogenic carcinoma (GCOC) based on the WHO classification of odontogenic tumors in 2005. The specimens of COC and 10 classic ameloblastoma (AB) were examined immunohistochemically to determine the expression of NF-kappaB p65, Ki-67 and MMP-9. NF-kappaB was mainly detected in the cytoplasm of most tumor cells, but was only detected in the nucleus of few tumor cells (rate of nuclear staining < 1%). The expression of Ki-67 was significantly higher in GCOC than in CCOT (P < 0.001), DGCT (P < 0.05) and AB (P < 0.005). MMP-9 was detected both in tumor cells and stromal cells. GCOC showed significantly higher percentage of MMP-9 positive cases in stromal cells than CCOT, DGCT and AB (P < 0.05). NF-kappaB may minimally affect the progression and invasion of COC. GCOC shows significantly higher proliferative activity and aggressiveness than CCOT and DGCT. MMP-9 in stroma may play a key role in the invasion of GCOC.

P.239 Gorlin cyst: A case treated with PRP and bone grafting

P.239 Gorlin cyst: A case treated with PRP and bone grafting

Investigation of the β-catenin gene in a case of dentinogenic ghost cell tumor

Dentinogenic ghost cell tumor (DGCT) is considered as a neoplastic counterpart of the calcifying odontogenic cyst (COC). beta-catenin mutations have been described in COC suggesting a critical role in its histogenesis. In this study, we report a patient with DGCT contains a missense mutation on codon 3 (ACT --> TCT) of beta-catenin gene. Immunohistochemistry showed nuclear, cytoplasmic, and membranous accumulation of beta-catenin in the tumor cells. TUNEL assay showed positive signals in nucleated cells adjacent to the ghost cells. Our data suggest that beta-catenin plays an important role in the tumorigenesis of DGCT. DGCT may develop by an improper differentiation process coordinated by Wnt signaling pathway. Further studies are needed to determine the genotypic/phenotypic characteristics of ghost cell-containing odontogenic lesions.

Calcifying odontogenic cyst: Radiologic findings in 11 cases

This retrospective study examines the relationship between histopathologically diagnosed cases of in calcifying odontogenic cysts (COCs) on the adjacent dentition. The records including diagnostic radiograph images of 11 patients treated for COCs from 1991 to 2004 were analyzed and correlations made between radiologic and histopathologic features. Special attention was applied to the associations between COCs and adjacent teeth. Radiologic and histopathologic features of the 11 lesions were variable, with some lesions being more solid and others more cystic. Calcifications varied from small flecks to solid calcified areas of frank odontoma. In 7 cases, the COC was associated with an impacted tooth and 5 of these cases involved COC-enveloping teeth. All cases excepting one in an edentulous jaw segment showed positional changes of adjacent teeth, including impaction. The radiographical features of COCs are varied but there is a high frequency of changes in adjacent teeth, including dental impaction. The presence of impaction or displacement of teeth for COCs occurring in the posterior segments of the jaw, and absence of this finding for anteriorly placed lesions may be indicative of the timing of COC development.