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Extraosseous calcifying odontogenic cyst: a case report and a literature review
Cazal, Claudia;Sobral, Ana Paula Veras;Silva, Vania Cavalcanti Ribeiro da;Araújo, Vera Cavalcanti de;
Jornal Brasileiro de Patologia e Medicina Laboratorial , 2005, DOI: 10.1590/S1676-24442005000600012
Abstract: the calcifying odontogenic cyst is an uncommon odontogenic lesion that can have intra- or extraosseous occurrence with both cystic or tumor behavior. a report of an extraosseous calcifying odontogenic cyst (ecoc) in a 57-year-old black woman is presented as well as a review of the literature about the lesion. the clinical, radiographic and histopathologic features are discussed, along with etiology and treatment.
Ameloblastomatous calcifying odontogenic cyst; a case report
M Seyed-majidi,K Nosrati,S Haghanifar
Journal of Isfahan Dental School , 2009,
Abstract: Introduction: 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.Case Report: In this report, we present a rare case of calcifying odontogenic cyst with ameloblastic proliferation, an extremely rare histologic variant, in a 15 year old female in the left mandibular molar region. The lesion was surgically removed. After enucleation and curettage, no recurrence recorded in next two years.Conclusion: Ameloblastomatous calcifying odontogenic cyst, microscopically resembles unicystic ameloblastoma except for the ghost cells and dystrophic calcifications within the proliferative epithelium. There has always been confusion about its nature as a cyst, neoplasm or hamartoma. Microscopically, this cyst shows ameloblastic proliferation in the connective tissue but no characteristics of basal cell hyperchromatism, vacuolization and nuclear polarization which is often seen in ameloblastoma, are present here. Ameloblastoma ex COC and ameloblastomatous COC are morphologically and clinically entirely different and easily distinguishable.Key words: Calcifying odontogenic cyst, Ghost cell, Gorlin Cyst, Ameloblastic proliferation
Predictive Factors of Potential Malignant Transformation in Recurrent Calcifying Cystic Odontogenic Tumor: Review of the Literature  [PDF]
Sepideh Mokhtari,Zhaleh Mohsenifar,Maedeh Ghorbanpour
Case Reports in Pathology , 2013, DOI: 10.1155/2013/853095
Abstract: Calcifying cystic odontogenic tumor (CCOT) demonstrates considerable diversity in histopathology and clinical behavior. Ghost cell odontogenic carcinoma (GCOC) is the rare malignant counterpart of CCOT and it frequently arises from malignant transformation of a recurrent CCOT. In this paper, we present a case of CCOT and discuss its distinct histopathologic features in recurrence. Then, we will have a review on clinical, histopathological, and immunohistochemical aspects of GCOC in the literature. Predictive factors of malignant transformation in a benign CCOT will also be discussed. 1. Introduction Calcifying odontogenic cyst (COC) represents 2% of all odontogenic lesions in the jaw [1]. It demonstrates considerable diversity in histopathology and clinical behavior. Latest subclassification of World Health Organization (WHO) has renamed this lesion as calcifying cystic odontogenic tumor (CCOT) [2]. Ghost cell odontogenic carcinoma (GCOC) is the malignant counterpart of this tumor and it frequently arises from malignant transformation of CCOT after multiple recurrences [3]. Here, we present a case of recurrent CCOT and discuss its distinct histopathologic features as potential predictive factors of future malignancy. We will also have a review on clinical, histopathological, and immunohistochemical characteristics of GCOC in the literature. 2. Case Presentation A 54-year-old male presented with swelling in the right side of mandible. He had a history of right first molar extraction 5 years ago with subsequent abscess formation and without any treatment. Radiographic examination revealed a multilocular radiolucent lesion (Figure 1). Root resorption of right mandibular canine and premolars was also obvious. Incisional biopsy revealed a benign cystic lesion with typical histologic features of calcifying odontogenic cyst (Figure 2). The lesion was excised and extensively curetted. Serial panoramic radiographs were taken in 2-week, 3-, 11-, 13-, and 18-month follow-up (Figures 3 and 4). Continuous healing process was seen in panoramic views. However, in all radiographs a nonhealing radiolucent area with progressive increase in size was evident. This area was apparent in the radiograph of 18-month follow-up as a well-defined radiolucent lesion. Clinical examination revealed a swelling in the right side of mandible measuring 4 × 3?cm. The surface skin was intact with no erythema or tenderness and the patient had no lymphadenopathy. In computed tomographic sections, buccal and lingual cortex perforations were evident. Tumor recurrence was confirmed by
Dentinogenic Ghost Cell Tumor of the Peripheral Variant Mimicking Epulis  [PDF]
Uddipan Kumar,Hitesh Vij,Ruchieka Vij,Jitin Kharbanda,IN Aparna,Raghu Radhakrishnan
International Journal of Dentistry , 2010, DOI: 10.1155/2010/519494
Abstract: Dentinogenic ghost cell tumor (DGCT) is an uncommon locally invasive odontogenic tumor regarded by many as a variant of calcifying odontogenic cyst. The peripheral variant of this clinical rarity appears as a well-circumscribed mass mimicking a nonspecific gingival enlargement. Microscopic appearance of odontogenic epithelium admixed with focal areas of dentinoid formation and sheets of ghost cells giving the definitive diagnosis of dentinogenic ghost cell tumor imply that microscopic examination is compulsory for any gingival mass. Van Gieson histochemical stain further confirmed the nature of dentinoid-like material. A complete workup of a case of peripheral dentinogenic ghost cell tumor is presented in this paper and the current concept as well as the appraisal of literature is presented. 1. Introduction Calcifying odontogenic cyst (COC) is a unique jaw lesion, first recognized as a distinct entity by Gorlin et al. [1] and hence the eponym Gorlin cyst. Praetorius et al. [2] classified them into the cystic type (Type I) and the solid type (Type II). The solid variant of COC (Type II) is rare and is designated as dentinogenic ghost cell tumor (DGCT), although the first description of the solid variant was given by Fejerskov and Krogh as calcifying ghost cell odontogenic tumor [3]. DGCT is characterized microscopically by ameloblastoma-like odontogenic epithelial proliferation, presence of ghost cells, and dentinoid-like material [4]. Due to its diverse histological picture, several terms have been used by different authors to describe this lesion such as dentinogenic ghost cell tumor [2], calcifying ghost cell odontogenic tumor [3], keratinizing ameloblastoma [5], cystic calcifying odontogenic tumor [6], peripheral odontogenic tumor with ghost cell keratinization [7], dentinoameloblastoma [8], ameloblastic dentinoma [9], epithelial odontogenic ghost cell tumour [10], and odontogenic ghost cell tumor [11]. The term DGCT is commonly used, and the peripheral variant of this neoplastic entity is rare; only few reports with clinical, radiographic, and histologic documentation can be found in the English literature. A report of peripheral dentinogenic ghost cell tumor (PDGCT) and characterization of dentinoid material using Van Gieson special stain for the confirmation adds a new dimension to the diagnosis of DGCT. 2. Case Report A 40-year-old male patient reported to the dental clinics at Manipal College of Dental Sciences, Manipal University, with a complaint of missing teeth. Clinical examination disclosed a swelling measuring about 5?mm in diameter in
Intraosseous calcifying cystic odontogenic tumor  [cached]
Kler Shikha,Palaskar Sangeeta,Shetty Vishwa,Bhushan Anju
Journal of Oral and Maxillofacial Pathology , 2009,
Abstract: The calcifying odontogenic cyst was first reported by Gorlin et al . in 1962. It had been classified as a neoplasm related to the odontogenic apparatus because of its histological complexity and morphological diversity until it was renamed as a calcifying cystic odontogenic tumor by the WHO, in 2005. Here we describe a case of mandibular calcifying cystic odontogenic tumor in a 75-year-old male, which was present since five years, with a history of occurrence after the extraction of teeth in the involved region. The lesion was surgically removed and a histopathological examination revealed a cystic tumor with predominance of ghost cells and some amount of dentinoid tissue.
Pilomatricoma: A dermal analog of calcifying odontogenic cyst  [cached]
Kaveri H,Punnya A
Indian Journal of Dental Research , 2008,
Abstract: 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.
Peripheral calcifying cystic odontogenic tumour of the maxillary gingiva
Ana Lima, Dárcio Kitakawa, Janete Almeida, Adriana Aigotti Brand?o, Ana Anbinder
BMC Research Notes , 2012, DOI: 10.1186/1756-0500-5-455
Abstract: Authors report a rare case of a peripheral calcifying cystic odontogenic tumor of the maxillary gingiva. A 39-year-old male patient presented with a fibrous mass on the attached buccal gingiva of the upper left cuspid teeth. It was 0.7-cm-diameter, painless and it was clinically diagnosed as a peripheral ossifying fibroma. After an excisional biopsy, the diagnosis was peripheric calcifying cystic odontogenic tumor. The patient was monitored for five years following the excision, and no recurrence was detected.All biopsy material must be sent for histological examination. If the histological examination of gingival lesions with innocuous appearance is not performed, the frequency of peripheral calcifying cystic odontogenic tumor and other peripheral odontogenic tumors may be underestimated.A calcifying cystic odontogenic tumor (CCOT) is an extremely rare benign cystic neoplasm that is characterized by an ameloblastoma-like epithelium and ghost cells that have the potential to undergo calcification [1]. Originally, CCOTs were referred to as calcifying odontogenic cysts (COC). The structure was first described by Gorlin in 1962 as a distinct entity and was therefore called Gorlin cyst [2]. COC was considered as a developmental odontogenic cyst in the jaw. In their first report, Gorlin et al. [2] considered this lesion to be a possible analogue of the cutaneous calcifying epithelioma of Malherbe (the pilomatrixoma). COC accounted for approximately 1% of jaw cysts. In 1981, Praetorius et al. [3] studied and reevaluated 16 cases of COC and proposed that the group actually contained two entities, a cyst and a neoplasm. Since then, neoplastic potential has been investigated.In 2005, the World Health Organization (WHO) designated Gorlin’s cyst as a tumor and described it as belonging to a group of related neoplasms, including the benign cystic-type (CCOT), the benign solid-type dentinogenic ghost cell tumor, and the malignant ghost cell odontogenic carcinoma [1]. The dentino
Calcifying Odontogenic Cyst with Complex Odontoma: Histological and Immunohistochemical Features
Nooshin Mohtasham,Amin Rahpeyma,Saeedeh Khajeh Ahmadi,Mohsen Merati
Journal of Dental Materials and Techniques , 2012,
Abstract: The calcifying odontogenic cyst (COC) is a rare odontogenic cyst. Only 2% of all odontogenic cysts and tumors are COC. COC associated with odontoma (COCaO) reported in 24% of COCs. COCaO presents a greater incidence in female, with a ratio of 2 to 1. The highest incidence of COCaO occurs during the second decade with a mean age of 16 years, most frequently occurring in the maxilla (61.5%). Here, we describe a classic case of COCaO of the maxillary incisor-canine region in 17-year-old girl, and discuss the clinicopathological features and immunohistochemical finding of this tumor.
A unique case of a calcifying cystic odontogenic tumor  [PDF]
Shoko Gamoh, Yukako Nakashima, Hironori Akiyama, Kimishige Shimizutani, Takuro Sanuki, Junichiro Kotani, Koji Yamada, Shosuke Morita, Kazuya Tominaga, Masahiro Wato, Akio Tanaka
Open Journal of Stomatology (OJST) , 2013, DOI: 10.4236/ojst.2013.36052
Abstract:

The calcifying odontogenic cyst was first reported by Gorlin et al. in 1962. At that time, it was classified as a cyst related to the odontogenic apparatus, although it was later renamed as a calcifying cystic odontogenic tumor by the WHO calcification in 2005 due to its histological complexity, morphological diversity and aggressive proliferation [2]. Here, we describe a case of a calcifying cystic odontogenic tumor in a 4- year-old boy. The lesion was surgically removed, and the histopathological examination revealed it to be a cystic tumor with ghost cells, a stellate reticulum and small amount of dentinoid tissue in the cystic wall.

Notch signaling and ghost cell fate in the calcifying cystig odontogenic tumor
CH Siar, T Kawakami, RR Buery, K Nakano, M Tomida, H Tsujigiwa, PP Han, H Nagatsuka, HK Ng
European Journal of Medical Research , 2011, DOI: 10.1186/2047-783x-16-11-501
Abstract: Notch signaling is an evolutionarily conserved mechanism that enables adjacent cells to adopt different fates [1]. The Drosophila Notch gene encodes a transmembrane receptor with a large extracellular domain carrying multiple epidermal growth factor-like repeats and a cytoplasmic domain required for signal transduction [1]. In vertebrates, there are four Notch receptor proteins (Notch1, Notch2, Notch3, and Notch4) and five membrane-bound ligand proteins (Delta1, Delta2, Delta4, Jagged1, and Jagged2) [1]. Signals exchanged between neighboring cells through binding of ligand with its cognate receptor initiates short range events including differentiation, proliferation, and apoptotic events at all stages of development, thus controlling organ formation and morphogenesis [1]. Deregulation of Notch signaling has been implicated in developmental abnormalities and neoplasias [2].Ghost cells are large pale anucleate cells with homogeneous pale eosinophilic cytoplasm and very pale to clear central areas instead of a basophilic nucleus [3]. 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]. The World Health Organization Classification of Head and Neck Tumors considered ghost cells as transitory squamous cells at various stages of d
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