Dentinogenic ghost cell tumor associated with odontoma: report of a rare case and review of literature.

Dentinogenic ghost cell tumor (DGCT) is an uncommon odontogenic tumor considered to be the solid, tumorous variant of calcifying odontogenic cyst. DGCT is characterized by islands of ameloblastoma-like epithelial cells that resemble the enamel organ, the presence of ghost cells, and dentinoid material. This article reports a rare case of dentinogenic ghost cell tumor associated with an odontoma in an adult patient, with a review of the literature. To the best of the authors' knowledge, there have only been four case reports of DGCT associated with odontoma, all of which occurred in children and adults younger than 30 years old.

Dentinogenic ghost cell tumor: a case report and review of the literature.

The purpose of this report is to highlight the presentation and review the clinical and histopathological features of DGCT. There have been 130 DCGT diagnoses reported in the literature. DGCT is part of the odontogenic ghost cell tumor family which also includes the calcifying odontogenic cyst (COC) and the ghost cell odontogenic carcinoma (GCOC). In June of 2018, a 48-year-old female presented with a painless, soft tissue growth overlying the right mandibular alveolar ridge. Further workup of the lesion included a panoramic radiograph and maxillofacial computed tomography (CT) which revealed a well-defined, multilocular lytic expansile radiolucency occupying the right mandibular body. An incisional biopsy of the right mandibular gingival mass was performed which revealed an unusual odontogenic neoplasm with mineralization and ghost cells. The patient was subsequently treated with excisional biopsy of the right mandibular lesion via enucleation and curettage. The specimen was sent to pathology and the tumor was found to have an epithelial neoplastic proliferation resembling that of ameloblastoma, accompanied by foci of ghost cells. Since mandibular bone was involved, a diagnosis of a benign central DGCT with extension into the overlying gingiva was rendered. She was successfully treated with excisional biopsy via enucleation and curettage and has no evidence of recurrence at three years post-operatively. DGCT can exhibit locally aggressive behavior and is characterized by ameloblastoma-like epithelial cells and the presence of dentinoid material and ghost cells.

Recurrent Dentinogenic Ghost Cell Tumor: A Case Report.

BACKGROUND Dentinogenic ghost cell tumor (DGCT) is a rare, locally invasive odontogenic neoplasm, considered as a solid variant of the calcifying odontogenic cyst (COC). DGCT accounts for only 2% to 14% of all COCs and less than 0.5% of all odontogenic tumors. It is characterized by an ameloblastomatous odontogenic epithelium and the presence of ghost cells and dentinoid material. CASE REPORT A 42-year-old male patient presented to the clinic with recurrent pain and swelling in the left maxilla. The patient had a similar presentation 3 years before, which had been managed by excisional biopsy and was misdiagnosed as unicystic ameloblastoma. Examination revealed a mild swelling in the left cheek with boney expansion on the posterior area of the left maxilla, with mobility in adjacent teeth. Radiographic evaluation revealed a large, well-demarcated radiolucent lesion with a sclerotic border involving the left maxilla and associated with root resorption in the adjacent teeth. An incisional biopsy was performed, and a diagnosis of DGCT was made. Segmental maxillectomy with safe margins was conducted, considering the aggressiveness of this pathological entity. The defect was reconstructed using the buccal fat pad. Histopathology examination confirmed the diagnosis of DGCT, which was characterized by ameloblastomatous odontogenic epithelium, dentinoid material, and ghost cells. One month follow-up revealed good healing of the surgical site. The patient was scheduled for regular follow-up. CONCLUSIONS This case reports the aggressiveness and high recurrence rates of DGCT. It is recommended to treat DGCT aggressively with safe margins and a long-term follow-up.

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.

Quiste inflamatorio con tejido calcificado: presentación de un caso clínico / Inflammatory cyst with calcified tissue: presentation of a clinical case

Objetivo: Describir un caso de quiste odontogénico in- flamatorio cuya presentación imagenológica no es la habitual. Caso clínico: Acude a la clínica profesional de Pato- logía Oral de la Universidad Andrés Bello una paciente de género femenino de 13 años, sin antecedentes mórbidos, con un hallazgo radiográfico de lesión mixta ubicada entre los premolares inferiores derechos. Es derivada al posgrado de cirugía oral y maxilofacial para que le realicen una biopsia excisional de la lesión con una hipótesis diagnóstica de tumor odontogénico adenomatoide. Una vez realizado el estudio histopatológico, se define la lesión como quiste odontogénico inflamatorio, que por las características clínicas-radiográficas podría corresponder con un quiste residual de un diente tem- poral. Debido a la variabilidad en la presentación clínica e ima- genológica de las lesiones quísticas maxilofaciales, el estudio anatomopatológico es imprescindible para un correcto diag- nóstico y tratamiento (AU)

Aim: To present a case of inflammatory odontogenic cyst with unusual imaging presentation. Clinical case: A 13-year-old female patient, with no history of morbidity, with a radiographic finding of a mixed lesion located between the lower right premolars. The patient visited the professional Oral Pathology clinic of the Andrés Bello University and was referred to postgraduate oral and maxillofacial surgery for an excisional biopsy of the lesion, with a diagnostic hypothesis of adenomatoid odontogenic tu- mor. After the histopathological study, the lesion was defined as an inflammatory odontogenic cyst, which, based on clini- cal-radiographic characteristics, could be a residual cyst of a primary tooth. Due to the variability in the clinical and imaging pres- entation of maxillofacial cystic lesions, anatomopathological study is essential for correct diagnosis and treatment (AU)

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.

Active decompression and distraction sugosteogenesis for the treatment of calcifying odontogenic cyst.

The calcifying odontogenic cyst is as a benign, rare developmental odontogenic cyst with a wide range of histologic characteristics. It may present along with other odontogenic pathologies such as odontoma, ameloblastoma, adenomatoid odontogenic tumor, ameloblastic fibroma, and ameloblastic fibro-odontoma. Clinically, it can be an either intra- or extraosseous painless swelling that can produce cortical expansion. It affects mostly the anterior area of the mandible. Radiographically, it appears as a well-circumscribed unilocular radiolucency containing flecks of indistinct radiopacities. In about one third of cases, an impacted tooth is associated. In this paper, we employ a patient with an enormous calcifying odontogenic cyst to review both the pathology and active decompression and distraction sugosteogenesis, a novel technique employed to treat odontogenic entities. This dual approach usually results in an accelerated bone healing (sugosteogenesis), partial removal of the cystic epithelium, thickening of the wall, and migration of chronic inflammatory cells which triggered epithelial modulation, ultimately causing a realignment in the biologic behavior of the lesion.

Ghost cell odontogenic carcinoma arising in the background of a calcifying odontogenic cyst.

Ghost cell odontogenic carcinoma (GCOC) is a rare malignant neoplasm, representing 3% of all ghost cell lesions of the jaws. They can arise de novo or from a pre-existing calcifying odontogenic cyst (COC) or dentinogenic ghost cell tumour (DGCT). A systematic review of the literature reported only 12 cases of a GCOC arising from a pre-existing COC. This report highlights an additional case of a GCOC arising from a pre-existing COC after 3 years in an adolescent male. The patient initially presented with a painless swelling of the right mandibular corpus. Panoramic radiographic examination showed an expansive unilocular radiolucent lesion. After 3 years, the radiographic features appeared more aggressive with increased expansion and cortical perforation. A wide surgical resection was performed, whereby the lesion was diagnosed as a GCOC. Due to the rarity of these malignant neoplasms, limited information is available regarding their biological behaviour. One-year follow-up revealed no clinical signs of recurrence.

Calcifying cystic odontogenic tumor associated with ameloblastoma - A rare histological variant.

Calcifying odontogenic cysts (COCs) represent a heterogeneous group of lesions that exhibits a variety of clinical, behavioral, and histological features. COC or Gorlin's cyst is now classified by the World Health Organization as a tumor. Calcifying cystic odontogenic tumor (CCOT) is an uncommon developmental odontogenic lesion that demonstrates histopathologic diversity. 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 16-year-old girl with involvement of mandibular posterior region, which is an extremely rare histologic variant.

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.

Peripheral Developing Odontoma or Peripheral Ameloblastic Fibro-Odontoma Erupting to Oral Cavity Case Report / Odontoma en Desarrollo Periférico o Fibro-Odontoma Ameloblástico Periférico Eruptando a Cavidad Oral Reporte de Caso

ABSTRACT: The appearance of mixed odontogenic tumors into the oral cavity is a rare event. It is considered that some mixed tumors are only a stage in the complete development of a hamartomatous formation such as ameloblastic fibroodontoma and odontoma. Both pathologies share in common cellular elements which at one point makes them indistinguishable from each other. We present the case of a 21 year old patient who showed a mandibular growth whose histological elements present characteristics of both pathologies. The treatment was surgical excision of the lesion. There were no complications or recurrences to periodic reevaluation.

RESUMEN: La aparición de tumores odontogénicos mixtos en la cavidad oral es un evento raro. Se considera que algunos tumores mixtos son solo una etapa en el desarrollo completo de una formación hamartomatosa como el fibro-odontoma ameloblástico y odontoma. Ambas patologías comparten elementos celulares comunes que en un punto los hacen indistinguibles entre sí. Presentamos el caso de un paciente de 21 años que mostró un crecimiento mandibular cuyos elementos histológicos presentan características de ambas patologías. El tratamiento fue la escisión quirúrgica de la lesión. No hubo complicaciones o recurrencias a la reevaluación periódica.

Calcifying odontogenic cyst, dentinogenic ghost cell tumor, and ghost cell odontogenic carcinoma: A systematic review.

BACKGROUND: The aim of this study was to integrate the available data published on calcifying odontogenic cyst (COC), dentinogenic ghost cell tumor (DGCT), and ghost cell odontogenic carcinomas (GCOCs) into a comprehensive analysis of their clinicoradiological features, treatment, and recurrence. MATERIALS AND METHODS: An electronic search with no publication date restriction was undertaken in October 2017 in the following databases: PubMed, Medline Ovid, Web of Science, and Scopus. Eligibility criteria included publications containing enough clinical, radiological, and histopathological information to confirm a definite diagnosis of these lesions. Data were evaluated descriptively. RESULTS: The literature review indicated a total of 234 publications reporting 367 COCs, 55 DGCTs and 44 GCOCs. These lesions have a predilection for Asian males. COCs mainly affect the mandible and patients in the second decade of life, DGCTs mostly affect the mandible and patients in the fourth decade of life, and GCOCs mostly affect the maxilla and patients in the fifth decade of life. CONCLUSION: Conservative surgery was the most common therapy for COCs and DGCTs, while radical surgery was most common for GCOCs. This study provides important and interesting data that could help clinicians and surgeons as well as oral and maxillofacial pathologists with the diagnosis and management of these lesions.

Tube Decompression for Staged Treatment of a Calcifying Odontogenic Cyst-A Case Report.

The calcifying odontogenic cyst is a rare developmental odontogenic lesion with a distinguishing cystic lining containing "ghost" epithelial cells. The variation in clinical, radiographic, and histologic findings makes the treatment decision difficult for these lesions. There are very few reports on treatment, with enucleation and curettage being the standard surgical method of choice for the cystic type and excision of the tumor for the solid type. A 2-stage surgical approach consisting of initial decompression with tube placement followed by a secondary procedure of enucleation and curettage is common for other large odontogenic cysts, such as dentigerous and odontogenic keratocysts. To the authors' knowledge, this is the first case managed with an intended 2-stage approach in the form of decompression followed by enucleation and curettage.

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.