Quiste odontogénico calcificante. Análisis de características clínicas e imagenológicas en reportes de casos / Calcifying odontogenic cyst. Analysis of clinical and imaging characteristics in case reports

Introducción: El quiste odontogénico calcificante (QOC) es un quiste odontogénico raro de los maxilares, que en un gran número de casos se asociaa otras patologías. Debido a esto, la presentación clínica e imagenológica puede variar. El objetivo del presente trabajo fue determinar las características clínicas e imagenológicas del QOC intraóseo, sin asociación a otras patologías, publicadas en reportes de casos.Material y métodos:Se realizó una búsqueda en PubMed, Scopus, Epistemonikos y Web of Science, de reportes de casos de QOC intraóseo, sin asociación a otras patologías, que tuvieran exámenes imagenológicos y diagnóstico histopatológico.Resultados:Se incluyeron 20 artículos, con 21 casos clínicos. El QOC sepresentó principalmente en la segunda década de vida, sexo masculino y zona posterior mandibular. El aumento de volumen estuvo presente en la mayoría de los casos. El dolor, alteraciones de mucosa y movilidad dentaria se observaron en un menor porcentaje. Radiográficamente se observó una lesión de densidad mixta, unilocular, de límites definidos y regulares, con expansión de corticales. La perforación, desplazamiento dentario y reabsorción radicular fueron poco comunes. El tamaño fue principalmente mayor a 3cm.Conclusiones:La información clínica e imagenológica fue escasa en todos los artículos incluidos. La mayoría de las características que no coinciden con lo reportado por la literatura podrían tener su explicación en el hecho de que los casos publicados suelen ser atípicos.En la presenterevisión se encontraron características del QOC diferentes a las reportadas, que son relevantes para el diagnóstico diferencial.(AU)

Introduction: Calcifying odontogenic cyst (COC) is a rare odontogenic cyst of the jaws, which in many cases is associated with other pathologies and can generate differences in terms of clinical and imaging presentation. The aim of this article was to determinate the clinical and imaging characteristics of intraosseous COC, without association to other pathologies, published in case reports.Material and methods:A search in PubMed, Scopus, Epistemonikos and Web of Science was performed, for case reports of intraosseous COC, without association to other pathologies, which had imaging examinations and histopathological diagnosis.Results:20 articles were included, with 21 clinical cases. COC was found to occur mainly in the second decade of life, male sex, and mandibular posterior area. Swelling was present in most cases. Pain, mucosal alterations, and dental mobility were observed in a lower percentage. Radiographically, a mixed density lesion, unilocular, with defined and regular limits, which generates cortical expansion was observed. Perforation, tooth displacement and root resorption were uncommon. The size was mainly larger than 3cm.Conclusion:Clinical and imaging information was found to be scarce in all articles included in this review. Most of the characteristics that did not coincide with what is reported by the literature could be explained by the fact that published cases are usually atypical. In this review, different COC characteristics than those previously reported were found, which are relevant for differential diagnosis.(AU)

Cystic Odontogenic Tumor with Features of Calcifying Odontogenic Cyst in 3 Cats and a Dog.

This case series describes the clinical appearance, radiographic appearance and histopathologic features of 4 patients (3 cats and 1 dog) with unique cystic oral lesions that are consistent with a diagnosis of calcifying odontogenic cyst (COC). COC is a rare odontogenic lesion in humans that has not been previously reported in companion animals. Historically, COC in humans has been classified as a benign cystic neoplasm and as a non-neoplastic odontogenic cyst. Current classification favors the latter. The cases in this series also have ambiguous features regarding classification of the lesion as an odontogenic cyst versus benign neoplasia.

Compound odontoma associated with a calcifying odontogenic cyst. Case report and systematic review.

The objectives of the present study were to comprehensively evaluate all the published cases on compound odontoma associated with calcifying odontogenic cyst (COaCOC) in the English literature and to describe the clinical, imaging and therapeutic variables for this condition. In August 2020, an electronic search of the PubMed / MEDLINE, Web of Science, ScienceDirect, Springer, and Scopus databases was carried out. The eligibility criteria included publications with enough information to confirm the diagnosis. Furthermore, we present a clinical case of a 16-year-old male patient with OCCaC, who was treated with enucleation, obtaining favorable and functional results. A total of 32 cases reported in the literature that met the inclusion and exclusion criteria, including ours, were analyzed and discussed. The mayority of the patients were women (n = 17) with an average age of 14.4 years, the maxilla was the most affected bone (n = 22) and the maxillary anterior region was the area with the highest number of cases (n = 18), the main clinical presentations were the volume increase (n = 14) and asymptomatic (n = 14). The choice treatment was enucleation (n = 26) and, in most cases, no recurrence was reported (n = 20). This study allows to update the characteristics of the OCCaC, giving an effective vision of how to treat this rare pathological association made up of two conditions that are completely different from each other.

Primordial odontogenic tumor occurred in the maxilla with unique calcifications and its crucial points for differential diagnosis.

Primordial odontogenic tumor (POT) is a newly classified, mixed epithelial and mesenchymal odontogenic tumor, with only 17 reported cases to date. Herein, we report a case of POT that occurred in the right maxilla of a 10-year-old boy and reveal unique features in comparison with those previously reported. Radiologically, the lesion presented as a well-defined, unilocular radiolucency with notable radiopaque foci on the periphery. Microscopically, the tumor was mainly composed of dental papilla-like myxoid fibrous connective tissue, largely surrounded by non-keratinized squamous epithelium with numerous calcified particles, and partly enclosed by inner enamel epithelium-like columnar cells and enamel organ-like structures accompanied with cuboidal and/or stellate reticulum-like cells. Immunohistochemically, the epithelium tested positive for cytokeratin 14 and 19. Moreover, amelogenin and ameloblastin, matrix proteins relating to enamel formation, were positive in the covering epithelium. The tumor was enucleated as a whole, and no recurrence was recorded thereafter. Although the presence of numerous calcified particles was unique, we diagnosed this lesion as POT based on the above-described features. Furthermore, we emphasize the importance of the differential diagnosis of POT and other odontogenic tumors that resemble corresponding tooth germ components.

Calcifying odontogenic cyst associated with the impacted third molar: a case report.

Calcifying odontogenic cyst (COC) is a benign, locally aggressive, slow-growing lesion. Its occurrence constitutes about 2% of all odontogenic cysts. The most frequent sign is painless, slow growing swelling. Radiographically it appears as a well-defined unilocular radiolucency. The microscopical features of lesion showed well-delineated cystic proliferation of odontogenic epithelium with ghost cells and fibrous connective tissue wall. In the present study, 42 years old man who has COC associated with the impacted third molar treated with enucleation is reported after 6 months follow-up.

Calcifying Odontogenic Cyst Associated With Dentigerous Cyst in a 15-Year-Old Girl.

Calcifying odontogenic cyst (COC) is a rare odontogenic cyst with ameloblastic epithelial lining containing clusters of ghost cells. COCs have been described in association with several odontogenic tumors, more commonly odontomas and rarely with dentigerous cyst (DC). In this article, we describe a case of COC associated with DC in a 15-year-old girl, who presented with a swelling on the right middle third of the face, producing facial asymmetry. Panoramic radiography showed a well-circumscribed, corticated, and unilocular radiolucency at the level of the right maxillary sinus, involving 2 unerupted premolars. The lesion was enucleated and histologically revealed a COC associated with DC, which presented mucous metaplasia. Immunohistochemical reactions were performed to better illustrate this rare synchronous occurrence of COC and DC, showing positivity for CK5, CK14, CK19, and p63 in both lesions. CK18 was negative in COC, and Bcl-2 was negative in DC. Periodic acid Schiff highlighted the mucous cells in the DC lining.

Calcifying odontogenic cysts.

INTRODUCTION: Calcifying odontogenic cysts (COC) represent 0.3-0.8% of all odontogenic cysts. We describe the finding and the treatment of a COC in a 67-year-old female. OBSERVATION: An asymptomatic well-limited radioluscent mandibular lesion was fortuitously discovered on the panoramic X-ray in the periapical region of teeth No. 33 and 34. Treatment consisted in enucleation and curettage. Histologic examination was in favour of a COC. At 6 years follow-up, X-ray control showed new bone formation and the patient was free of symptoms. Pulp vitality was maintained in all teeth in the operated area. DISCUSSION: Total enucleation is the preferred treatment of COC. Absence of recurrence is attested by X-ray controls and pulp vitality tests.

Ghost Cell Tumors.

Ghost cell tumors are a family of lesions that range in presentation from cyst to solid neoplasm and in behavior from benign to locally aggressive or metastatic. All are characterized by the presence of ameloblastic epithelium, ghost cells, and calcifications. This report presents the cases of a 14-year-old girl with a calcifying cystic odontogenic tumor (CCOT) and a 65-year-old woman with a peripheral dentinogenic ghost cell tumor (DGCT) with dysplastic changes, a rare locally invasive tumor of odontogenic epithelium. The first patient presented with a 1-year history of slowly progressing pain and swelling at the left body of the mandible. Initial panoramic radiograph displayed a mixed radiolucent and radiopaque lesion. An incisional biopsy yielded a diagnosis of CCOT. Decompression of the mass was completed; after 3 months, it was enucleated and immediately grafted with bone harvested from the anterior iliac crest. The second patient presented with a 3-month history of slowly progressing pain and swelling at the left body of the mandible. Initial panoramic radiograph depicted a mixed radiolucent and radiopaque lesion with saucerization of the buccal mandibular cortex. An incisional biopsy examination suggested a diagnosis of DGCT because of the presence of ghost cells, dentinoid, and islands of ameloblastic epithelium. Excision of the mass with peripheral ostectomy was completed. At 6 and 12 months of follow-up, no evidence of recurrence was noted.

Role of hypoxia-related proteins in invasion of ameloblastoma cells: crosstalk between NOTCH1, hypoxia-inducible factor 1α, a disintegrin and metalloproteinase 12, and heparin-binding epidermal growth factor.

AIMS: Ameloblastoma AME is a benign tumour characterized by local invasiveness, high recurrence rates, and diverse histological patterns. The oxygen concentration is reduced in specific areas of the tumour microenvironment, which leads to intratumoral hypoxia. Crosstalk between NOTCH1, a disintegrin and metalloproteinase 12 (ADAM-12), hypoxia-inducible factor 1α (HIF-1α) and heparin-binding epidermal growth factor (HB-EGF) under hypoxic conditions has been implicated in invadopodia formation, tumour invasiveness, and metastasis development. The aim of this study was to analyse the expression of these proteins, in order to further elucidate the mechanisms underlying AME invasiveness. METHODS AND RESULTS: Twenty cases of AME, eight calcifying cystic odontogenic tumours CCOTs and 10 samples of dental follicle were used to investigate the expression of these proteins by immunohistochemistry with the primary antibodies anti-NOTCH1, anti-ADAM-12, anti-HIF-1α, and anti-HB-EGF. Immunostaining results were expressed as the percentage of stained area in images acquired in an AxioScope microscope equipped with an AxioCamHRc camera and a × 40 objective. The results showed that immunoexpression of all proteins was higher in the AME samples than in the CCOT and dental follicle samples (P < 0.05). CONCLUSIONS: AME showed an increased presence of proteins associated with tumour invasiveness, which indicates a possible role of these proteins in the biological behaviour of this tumour.

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.

[The glandular odontogenic cyst--a rare entity]. / Glanduläre odontogene Zyste - eine seltene Entität.

In this short communication, the very rare glandular odontogenic cyst (GOC) is presented as an independent entity. The GOC is a jawbone cyst of the maxilla and mandible. The typical radiological and histopathological characteristics of the GOC are described. Furthermore, differential diagnoses, current treatment options and the recurrence rates of this of entity are discussed.

Ameloblastic fibrodentinoma: report of a rare case.

AIM: This is to report a rare case of ameloblastic fibrodentinoma (AFD), including detailed history, histopathologic as well as radiographic findings. Her condition, a mixed odontogenic tumor, has rarely been reported in publications. We also included the gist of theories put forward regarding the histogenesis of this lesion. BACKGROUND: AFD is a rare and controversial entity; it boasts a variety of classifications and terminology based on its unique biological nature as well as histopathological features. CASE DESCRIPTION: The patient was a 13-year-old girl with a chief complaint of central unerupted tooth on the left mandible. Cone beam computed tomographic images revealed a mixed pericoronal and multilocular lesion surrounding the crown of the aforementioned tooth, displacing it apically to a large extent. CONCLUSION: It is important to differentiate AFD from other benign mixed odontogenic tumors with similar radiographic appearance because of different therapeutic approaches in some of these tumors. CLINICAL SIGNIFICANCE: From the clinical point of view, it is of paramount significance for dental practitioners to assess any delayed tooth eruption (more than 6 months and in comparison with its counterpart on the opposite side) radiographically to ascertain or rule out any such similar lesions.

Orthokeratinised odontogenic cyst mimicking periapical cyst.

Orthokeratinised odontogenic cyst (OOC) denotes the odontogenic cyst that microscopically has an orthokeratinised epithelial lining. OOC is characterised by a less-aggressive behaviour and a low rate of recurrence. This report describes a case of OOC involving posterior part of the mandible that mimicked periapical cyst in a 14-year-old boy. The initial clinical diagnosis was given as periapical cyst based on the clinical and radiographical features. Enucleation of the cyst was performed and the specimen was sent for histopathological examination. A definite diagnosis of OOC was made by histopathological examination of the biopsy specimen. This case emphases on including OOC in the differential diagnosis of radiolucencies occurring in the periapical region of non-vital tooth.

A rare case of recurring calcifying epithelial odontogenic cyst in the maxillary sinus: a case report and literature review.

Calcifying epithelial odontogenic cyst (CEOC) is an odontogenic cyst with epithelial lining. CEOC is a rare entity that occurs in a wide age range, does not show any gender predilection, and accounts for only 1% of all jaw cysts. The lesion generally occurs in the region anterior to maxillary and mandibular molars and either intraosseously or extraosseusly. This entity might present as a cystic or solid lesion. Enucleation is the recommended treatment for a simple, unicystic CEOC. A case of recurring CEOC in the right maxilla antrum is presented here. The patient presented to the authors after postsurgical recurrence. The case was evaluated thoroughly, and the cyst was resolved.

Ghost cell differentiation and calcification in ameloblastic fibroma.

Ghost cell differentiation within an ameloblastic fibroma is extremely rare. The ghost cells found in an ameloblastic fibroma in previously reported cases were all associated with a typical calcifying odontogenic cyst. Here, we report a case of an ameloblastic fibroma with focal ghost cells and calcifications in some neoplastic epithelial islands, but without other histologic manifestations consistent with a calcifying odontogenic cyst. The patient was a 13-year-old Chinese boy who presented with a bony-hard swelling in the posterior mandibular region over a 6-month period. Radiographs showed a well-defined multilocular radiolucency associated with an unerupted tooth. The lesion was mostly cystic-solid and comprised of odontogenic epithelial strands, islands and myxoid ectomesenchymal component microscopically. Small groups of ghost cells and calcification were noted in the epithelial islands.

Calcifying cystic odontogenic tumor radiographically mimicking a sinus mucocele.

The calcifying cystic odontogenic tumor or Gorlin cyst is an uncommon lesion with a variable clinical behavior and considerable histopathologic diversity. The authors report a case of calcifying cystic odontogenic tumor that was being treated as a maxillary sinus mucocele. The possibility of mimicking numerous odontogenic and nonodontogenic lesions makes the calcifying cystic odontogenic tumor difficult for a clinical diagnosis. The present case demonstrates that a specific knowledge in oral pathology is required to differentiate odontogenic lesions.