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.

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.

[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.

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.

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.

Calcifying odontogenic cyst: a rare report of a nonneoplastic variant associated with cholesterol granuloma.

AIM: To report a case of a non-neoplastic variant of calcifying odontogenic cyst (COC) with the lining epithelium showing ameloblastomatous proliferation and capsule exhibiting features of a cholesterol granuloma. The importance of delineating this histologic variant from unicystic ameloblastoma and the formation of cholesterol granuloma in this variant is discussed. BACKGROUND: Calcifying odontogenic cyst is a developmental jaw cyst, which presents itself as both the neoplastic and the non-neoplastic forms. The ameloblastomatous variant of COC is often mistaken for unicystic ameloblastoma and treated aggressively. CASE REPORT: A 68-year-old female who presented with a cystic enlargement of the posterior mandible on the right side was suggestive of unicystic ameloblastoma based on radiography and initial biopsy report. Microscopic examination of the excision specimen, however, was fitting in favor of calcifying odontogenic cyst with ameloblastomatous proliferation. CONCLUSION: Identifying the non-neoplastic ameloblastomatous variant of COC from a cystic ameloblastoma is crucial as the treatment of the two lesions vary considerably. CLINICAL SIGNIFICANCE: This case emphasizes the need for thorough examination of the entire surgical specimen before arriving at an appropriate diagnosis.

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.

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.

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.

Rare hybrid odontogenic tumor in a 2-year-old child.

Hybrid odontogenic tumors are rare conditions that can affect the oral maxillofacial region and usually occur in adults as an asymptomatic swelling. Hybrid odontogenic tumors exclusively involving adenomatoid odontogenic tumor (AOT) and calcifying cystic odontogenic tumor (CCOT) are rare, with only 4 reported cases. In addition, there are only few studies describing the presence of abortive enamel in AOT and, to our knowledge, CCOT was not present in any of them. We described a rare case of AOT associated with CCOT and abortive enamel formation in a 2-year-old child, a condition not well described in the international literature. Secretory cell activity was assessed by periodic acid-Schiff and Congo red stains.

Importance of cone beam computed tomography for diagnosis of calcifying cystic odontogenic tumour associated to odontoma. Report of a case.

The calcifying cystic odontogenic tumour (CCOT) is a rare benign cystic neoplasm not infrequently associated with odontoma. This report documents a case of CCOT associated with compound odontoma arising in the anterior maxilla in a 25-year-old woman. Conventional radiographs showed a large calcified mass with poorly visualized radiolucent margins. The extent and condition of the internal structure of the CCOT associated with odontoma was able to be determined based on radiographic findings from cone beam computed tomography. This advanced image technique proved to be extremely useful in the radiographic assessment of this particular neoplasm of the jawbones.

[Mixed odontogenic tumours]. / Tumeurs odontogéniques mixtes.

Mixed odontogenic tumors include all tumors of the teeth both epithelial and mesenchymatous. Most of these tumors are rare (ameloblastic fibroma, ameloblastic fibro-odontoma and fibrodentinoma, odontoameloblastoma, calcified odontogenic cyst). Other tumors such as odontoma are frequent. They have in common a benign aspect, their often-fortuitous observation, a higher rate in the young adult and surgical treatment by complete exeresis preventing recurrence. They present a radiolucent lacuna but appear different depending on the presence or not of intralesional calcifications revealing the production of more or less mature dental tissue. The diagnosis may be confirmed only by an anatomopathological examination, along with clinical and radiological observations.

Peripheral calcifying odontogenic cyst.

Calcifying odontogenic cyst is a rare lesion representing about 1% of jaw cysts. It may occur in a central (intraosseous) or peripheral (extraosseous) location. A case of peripheral calcifying odontogenic cyst located on the gingiva, appearing as a painless, circumscribed, sessile, and pink to red nodule has been reported.Peripheral, in contrast to central, calcifying odontogenic cyst tends to affect older patients. Peripheral calcifying odontogenic cyst is a less aggressive lesion than the central counterpart, and a simple excision biopsy is curative. The histologic findings of an epithelium rich in ghost cells has helped in making the diagnosis.

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.