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.     

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.     

Relative frequency of peripheral odontogenic tumors: a study of 45 new cases and comparison with studies from the literature

BACKGROUND: Peripheral (extraosseous) odontogenic tumors are rare, and reports in the literature have mainly been single case reports or a small series of cases. The aim of this study was to determine the relative frequency of peripheral (extraosseous) odontogenic tumors relative to one another and relative to their central (intraosseous) counterparts in an oral pathology biopsy service and to compare these data with information available in the literature. METHODS: The files of the Pacific Oral and Maxillofacial Pathology Laboratory of the University of the Pacific, San Francisco, CA, USA, served as the source of material for this study. Files were systematically searched for all cases of peripheral odontogenic tumors (POTs) during a 20-year-period. RESULTS: There were 91,178 cases accessed in which central and POTs were identified in 1,133 (1.24%), central tumors in 1,088 (1.2%), and peripheral tumors in 45 (0.05%). Peripheral tumors accounted for 4% of all 1133 central and POTs. Peripheral odontogenic fibroma (PODF) was the most common of the 45 POTs accounting for 51.1% (23 cases) followed by peripheral ameloblastoma (PA) 28.9% (13 cases) and peripheral calcifying cystic odontogenic tumor (PCCOT) 13.3% (six cases). Peripheral calcifying epithelial odontogenic tumor, peripheral ameloblastic fibroma, and peripheral ameloblastic carcinoma were also identified--each comprised 2.2% (one case each). PODF was more common than its central counterpart by a 1.4:1 ratio. This was the only peripheral tumor that was more common than its central counterpart. PA accounted for 9.3% of all ameloblastomas and PCCOT for 26% of all calcifying cystic odontogenic tumors. CONCLUSION: There is only scarce information in the literature on the relative frequency of POTs. Additional studies should be conducted to determine the true relative frequency. To ensure accuracy, pathologists with experience in the field of odontogenic tumors should conduct these studies. Intraosseous tumors that perforate through the bone to the gingival tissue, clinically presenting as 'peripheral tumors' should be excluded.     

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

Toida M: So-called calcifying odontogenic cyst: review and discussion on the terminology and classification. J Oral Pathol Med 1998; 27: 49–52. © Munks-gaard, 1998.
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.     

Bilateral Calcifying Odontogenic Cyst: A Rare Entity

The calcifying odontogenic cyst (COC) accounts for 1 % of jaw cysts, found most commonly as central lesion. The COC usually manifests itself as a painless, slow growing swelling, anterior to the first molar region. Radiographically it appears as a well-defined, unilocular radiolucency, and may have a radiopaque mass in its center. Majority of the cases appear before the fourth decade of life. The COC is found rarely in patients in the first decade of life. Histopathological features include a cystic lining demonstrating characteristic “Ghost” epithelial cells with a propensity to calcify. Here we report the unusual occurrence of a case of 8 year child diagnosed with bilateral COC on right and left side of mandible.     

Central odontogenic fibroma current concepts

The author reviews current knowledge concerning the central odontogenic fibroma, which at present is incompletely understood, and reaches the following conclusions. 1) The separation of this lesion into simple and WHO types remains valid because they exhibit different histologic features. However, more care should be taken in rendering the diagnosis of the WHO type than in the past; unlike the simple type, it is a fibroblastic lesion. 2) Complex central odontogenic fibroma is a more appropriate term than the WHO type because the WHO does not use the latter term in its 1992 manual. 3) The microscopic distinction of simple odontogenic fibroma from desmoplastic fibroma remains difficult in some cases. 4) The granular cell odontogenic tumor, which has sometimes been referred to as a type of odontogenic fibroma, is a separate entity, although some simple odontogenic fibromas exhibit scattered granular cells. 5) The separation of lesions that have been reported recently as odontogenic fibromas with giant cell reactions from central giant cell granulomas that exhibit foci of odontogenic epithelium requires further study.     

Peripheral ameloblastoma of the gingiva: the importance of diagnosis

BACKGROUND: Peripheral ameloblastoma is an extremely rare epithelial odontogenic tumor, limited to the soft tissues of the gums or oral mucosa. Although the lesion is benign, it may be locally aggressive. METHODS: The present study describes the case of a 31-year-old male presenting a firm, symptomless tumor mass of irregular appearance and measuring approximately 12 mm in diameter, located in the distal zone of 4.7. RESULTS: An excision biopsy was performed. The lesion was covered with hyperplastic squamous epithelium, with islets of epithelial cells located at subepithelial level. The cells in the peripheral zone adopted a palisade distribution, and presented the appearance of a lax reticulum at central level. A fibroblastic stroma was observed between the islets. The diagnosis was peripheral ameloblastoma. CONCLUSIONS: Although the origin of the lesion remains unclear, it is able to recur and undergo malignant transformation. Consequently, peripheral ameloblastoma should not be viewed as a harmless mass.     

Epulides in the dog: a review

This article is based on a review of the literature and the study of pathology sections obtained from various veterinary pathology laboratories. Epulis is a non-specific, clinical designation for a localized, exophytic growth on the gingiva. Four reactive epulides occur in human beings, namely focal fibrous hyperplasia (fibrous epulis). pyogenic granuloma. peripheral giant cell granuloma (giant cell epulis. and peripheral ossifying fibroma (calcifying fibrous epulisl). The first three also occur in dogs but only focal fibrous hyperplasia appears to be common. The peripheral ossifying fibroma has not yet been reported in dogs. Odontogenic tumors occurring on the gingiva (i.e., as epulides) are referred to as peripheral odontogenic tumors. Three types have been reported in dogs. One, the common fibromatous epulis. is equivalent to the rare peripheral odontogenic fibroma in human beings. Another, the acanthomatous epulis. appears to be a form of ameloblastoma but differs from the peripheral ameloblastoma in human beings in that it invades bone; its biological behavior is therefore that of the human intraosseous ameloblastoma. The third, a rare lesion, has been referred to in the veterinary literature as a calcifying epithelial odontogenic tumor, although it is not the canine counterpart of the human CEOT The term, amyloid-producing odontogenic tumor , has been suggested as being appropriate for this lesion.     

Unusual peripheral odontogenic tumors in the differential diagnosis of gingival swellings

Differential diagnosis of gingival mass lesions includes several conditions and causes. Peripheral odontogenic tumors may mimic gingival swellings and, although rare, must be included in the differential diagnosis. The purpose of this article is to describe 3 different cases of peripheral odontogenic tumors and to discuss the differential diagnosis of gingival swelling. Histologic examination is mandatory when localized gingival swellings are surgically removed.     

10-Year Follow-up of Calcifying Odontogenic Cyst in the Periapical Region of Vital Maxillary Central Incisor

Introduction

Radiographic images may lead to misinterpretations of lesions of endodontic and nonendodontic origin. This report describes a case of a 10-year follow-up of a calcifying odontogenic cyst (COC) in the periapical region of a vital maxillary central incisor in a 9-year-old boy.

Methods

The patient revealed a history of a swelling in the periapical area of tooth #9. The patient denied any dental trauma or history of pain. Clinical examination revealed no mobility, but there was discrete discomfort when horizontal pressure was applied. Pulp vitality was present in all maxillary anterior teeth. Radiographs revealed an oval radiolucent lesion in the periapical region of maxillary central incisor. The therapeutic option was enucleation of the periapical lesion and histologic examination of the specimen. Microscopic findings suggested the diagnosis of a COC.

Results

At a follow-up visit 10 years after surgery, panoramic and periapical radiographs showed new bone formation; the patient did not have any pain, and pulp vitality was maintained in all teeth in this area.

Conclusions

A COC should be part of the differential diagnosis of other jaw lesions, such as apical periodontitis. The definitive diagnosis of a COC can only be made after microscopic evaluation of the specimen. The follow-up is a helpful reference because it confirms the survival of pulp tissue and no recurrence of the COC.     

Peripheral calcifying cystic odontogenic tumour and peripheral dentinogenic ghost cell tumour: an updated systematic review of 117 cases reported in the literature

Purpose: To integrate the available data published on peripheral calcifying cystic odontogenic tumour (CCOT) and peripheral dentinogenic ghost cell tumour (DGCT) into a comprehensive analysis of its clinical and radiologic features.

Methods: An electronic search was undertaken in May, 2016. Eligibility criteria included publications reporting cases of peripheral CCOTs/DGCTs having enough clinical, radiological and histological information to confirm a definite diagnosis. Demographic data, lesion site and size, treatment approach and recurrence were analyzed.

Results: Hundred and thirty-eight lesions were found (65 publications), and 117 lesions (63 publications) with enough information were analyzed (55 CCOTs, 50 DGCTs, 12 unknown). Mean age of patients was 51.3?±?23.4 (min–max, 1–92), with higher mean age for the DGCTs variant. The lesions were more prevalent in the mandible, anterior region of the jaws, and in the second, sixth and eighth decades, with an equal sexual distribution. About 20% of all lesions showed signs of erosion of the underlying bone, with a higher rate for DGCTs. The mean lesion size was 1.3?±?0.8 (min–max, 0.4–3.0). Time of follow-up was informed for 37 lesions, with a mean?±?SD of 30.2?±?21.0 months (min–max, 6–84). Almost all lesions were treated by conservative surgery; only three recurrences were reported.

Conclusions: Peripheral CCOTs/DGCTs are rare lesions. Most of the lesions were treated by simple excision with or without curettage of the underlying bone. As the recurrence rate is very low, a conservative approach seems to be enough for the great majority of cases.     

Ameloblastomas in the horse: a critical review and report of an additional example

Previously published cases of ameloblastoma in the horse are reviewed in detail for their acceptability as examples of that tumor; an additional one is described. So far, this rare equine lesion has been shown to have two histologic patterns. The first consists of islands and sheets of epithelium that exhibit the basal cell characteristics of ameloblastoma; the central cells comprise stellate reticulum. The second exhibits these basal cell features less markedly and the central cells are spindle-shaped and closely packed. The biologic behavior of the equine ameloblastoma is thought to be the same as in human beings, a slowly growing, invasive lesion that does not metastasize. However, this conclusion requires confirmation.     

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提要：外周性牙源性肿瘤又称骨外型牙源性肿瘤或软组织牙源性肿瘤,主要发生在牙龈,包括牙源性真性肿瘤及错构瘤。外周性牙源性肿瘤约占所有牙源性肿瘤的4%左右,英文文献表明其中外周性牙源性纤维瘤最多见,其次是外周性成釉细胞瘤及外周性牙源性钙化囊性瘤。外周性牙源性肿瘤临床上容易与牙龈发生的炎症性或反应性病变相混淆,明确诊断依赖组织病理学检查。外周性牙源性肿瘤不包括骨内型牙源性肿瘤穿破骨皮质侵犯牙龈。外周性牙源性肿瘤预后普遍好于相应的骨内型肿瘤,但切除不彻底仍可复发,建议长期随访。     

Peripheral dentinogenic ghost cell tumor

A case of dentinogenic ghost cell tumor, that has originated peripherally in the jaw, is presented and the literature reviewed with particular reference to the origin of the tumor. The total number of central and peripheral cases reported in the English literature is 10 and although mucosal infiltration is common, peripheral origin of the neoplasm could be verified in only 3 cases.     

Cementoblastoma and osteoblastoma: a comparison of histologic features

Three cases of cementoblastoma (CB) and three cases of osteoblastoma (OB) were histologically compared. The aim of the study was to investigate whether CB and OB are different in other aspects than being connected with a tooth. CB cases were the following: maxillary lesions in a 23-year-old woman and a 22-yr-old man and a mandibular lesion in a 28-yr-old man. In one case the tumor was fused to the roots of two teeth. Of the OB cases, one occurred in the mandible of a 27-yr-old man, one in the ankle of a 19-yr-old male and one in the thoracic vertebrae of a 27-yr-old man. Histologically, CB and OB had the same appearance including peripheral spiculae rimmed by swollen blasts. This histologic similarity between OB and CB indicates that the diagnosis CB should not be made unless the lesion is connected with a tooth.     

Persistent apical periodontitis associated with a calcifying odontogenic cyst

Aim  To report a case of calcifying odontogenic cyst (COC) that was suggestive of apical periodontitis adjacent to the roots of the maxillary incisor teeth.
Summary  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.
Key learning points  • 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.     

Immunohistochemical demonstration of enamel proteins in odontogenic tumors

Immunohistochemical localization of two enamel proteins, amelogenin and enamelin, in comparison with that of keratin, was determined in odontogenic tumors and the allied lesions in order to verify functional differentiation of the tumor cells as ameloblasts. Amelogenin and enamelin were demonstrated in small mineralized foci and in the tumor cells surrounding them in adenomatoid odontogenic tumor (AOT), calcifying epithelial odontogenic tumor (CEOT), and calcifying odontogenic cyst (COC). Hyaline droplets in AOT showed positive staining for both enamel proteins. These mineralized and hyaline materials were not positive for keratin, although tumor cells were positive. On the other hand, no immunoreaction for enamel proteins was obtained in ameloblastoima and odontogenic epithelial cell nests within myxoma and epulis. The results suggest that tumor cells of AOT and CEOT and lining epithelial cells of COC show ameloblastic differentiation in part, but that ameloblastoma cells do not attain functional matauration as secretory phase ameloblasts.     

The relationship of adamantinomatous craniopharyngioma to ghost cell ameloblastoma of the jaws: a histopathologic and immunohistochemical study

The objective of this investigation was to study the relationship of the ghost cell ameloblastoma (GCA). which is a form of type II calcifying odontogenic cyst (COO, to the adamantinomatous craniopharyngioma (ACP). H&E sections of 26 examples of ACP were compared to three cases of GCA and to the reported microscopic features of that tumor. Clinical records of the ACPs were studied to determine their biologic behavior compared to that of the ameloblastomas. Immunohistochemical studies of nine examples of ACP were performed for KL1 (high mol.wt cytokeratins), 5D3 (low mol.wt cytokeratins) and involucrin (characteristic of terminally differentiated keratinocytes) using the peroxidase–amiperoxi–dase method. The results were compared with those reported for COC and ameloblastoma. ACP and GCA exhibited similar microscopic features, including pre–ameloblasts, tissue resembling stellate reliculum, ghost cells and calcifications; both tumors grew slowly and were invasive. ACP and COC. and by interpolation GCA. exhibited similar features with all three antibodies. The ghost cells did not exhibit any immunoreactivity but the adjacent cells stained positively for involucrin. The immunological features of ACP were similar to those reported in ameloblastomas for squarnous differentiation. However, because of their rarity, no ameloblastomas exhibiting keratinization. including ghost cells, have yet been studied with these antibodies. We conclude that ACP and GCA are homologous lesions.     

Squamous odontogenic tumor and squamous odontogenic tumor-like proliferations in odontogenic cysts: An updated analysis of 170 cases reported in the literature

Purpose

To integrate the available data published on squamous odontogenic tumors (SOT) and squamous odontogenic tumor-like proliferations in odontogenic cysts (SOT-LPOC) into a comprehensive analysis of their clinical/radiologic features.

Histopathologic study of epithelial components in the connective tissue wall of unilocular type of calcifying odontogenic cyst

Histopathologic study of satellite cysts and odontogenic epithelial islands in connective tissue wall of unilocular type of calcifying odontogenic cyst (COC) was made. The material was 13 cases consisting of 3 simple unicystic COCs, 9 odontome producing COCs and 1 ameloblastomatous proliferating COC. Satellite cysts were found in 6 cases, and were histologically classified into following types: simple cystic, odontome producing and ameloblastomatous. Histologic types of satellite cysts did not coincide with those of main cystic lesions in some cases. Odontogenic epithelial islands with or without proliferating feature were found in 9 cases, and were found in all cases with satellite cysts. Melanin and melanocytes were seen in an ameloblastomatous satellite cysts of 1 of 3 pigmented COCs.