RESUMO
Calcifying odontogenic cyst (COC), once called calcifying cystic odontogenic tumor, is classified under the category of odontogenic cysts. However, the proliferative capacity of the lesional epithelium and consistent nuclear ß-catenin expression raise questions about its current classification. This study aimed to determine whether COC would be better classified as a neoplasm in the histologic and molecular context. Eleven odontogenic lesions diagnosed as COC or calcifying cystic odontogenic tumor were included in this study. The growth patterns of the lesional epithelium were analyzed histologically in all cases. ß-catenin immunohistochemistry and molecular profiling using Sanger sequencing and whole-exome sequencing were performed in 10 cases. Of the 11 cases studied, histologic features reminiscent of so-called adenoid ameloblastoma were observed in 72.7% (8/11), and small islands of clear cells extended into the wall in 36.4% (4/11). Intraluminal and/or mural epithelial proliferation was found in 72.7% of the cases (8/11). Nuclear ß-catenin expression was observed focally in all 10 cases studied, mainly highlighting epithelial cells forming morules and adjacent to dentinoid. CTNNB1 hotspot mutations were detected in 60.0% of the cases (6/10). All the remaining cases had frameshift mutations in tumor-suppressor genes involved in the WNT pathway, including APC and NEDD4L. Recurrent WNT pathway mutations leading to nuclear translocation of ß-catenin and distinct epithelial growth patterns found in COC are the neoplastic features shared by its solid counterpart, dentinogenic ghost cell tumor, supporting its classification as a tumor rather than a cyst.
Assuntos
Mutação , Cisto Odontogênico Calcificante , Via de Sinalização Wnt , Humanos , Feminino , Masculino , Cisto Odontogênico Calcificante/patologia , Cisto Odontogênico Calcificante/genética , Adulto , Via de Sinalização Wnt/genética , Pessoa de Meia-Idade , beta Catenina/genética , beta Catenina/metabolismo , Ameloblastoma/genética , Ameloblastoma/patologia , Ameloblastoma/metabolismo , Adolescente , Adulto Jovem , Neoplasias Maxilomandibulares/genética , Neoplasias Maxilomandibulares/patologia , Biomarcadores Tumorais/genética , Biomarcadores Tumorais/análise , Tumores Odontogênicos/genética , Tumores Odontogênicos/patologia , Idoso , CriançaRESUMO
OBJECTIVE: To analyze the three-dimensional radiographic characteristics of calcifying odontogenic cyst and calcifying epithelial odontogenic tumor using spiral computed tomography (CT) and cone-beam computed tomography (CBCT). METHODS: Clinical records, histopathological reports, and CBCT or non-enhanced spiral CT images of 19 consecutive patients with calcifying odontogenic cyst (COC) and 16 consecutive patients with calcifying epithelial odontogenic tumor (CEOT) were retrospectively acquired, and radiographic features, including location, size, expansion, internal structure and calcification, were analyzed. RESULTS: Among the 19 COC cases (12 males and 7 females, with an average age of 27 years), 89.5% (17/19) of the lesions originated from the anterior and premolar areas, 100.0% of them exhibited cortex expansion, and 78.9% had discontinued cortex. Among the 16 CEOT cases (3 males and 13 females, with an average age of 36 years), 81.3% (13/16) of the lesions were in the premolar and molar areas, 56.3% of them exhibited cortex expansion, and 96.8% had discontinued cortex. According to the distribution of internal calcifications, these lesions were divided into: â (non-calcification type): absence of calcification; â ¡ (eccentric marginal type): multiple calcifications scattered along one side of the lesion; â ¢ (diffused type): numerous calcifications diffusely distributed into the lesion; â £ (plaque type): with a ≥ 5 mm calcified patch; â ¤ (peri-coronal type): multiple calcifications clustered around impacted teeth. Calcifications were present in 73.7% of COC lesions, including 9 type â ¡, 3 type â ¢ and 2 type â £ lesions, and 42.8% of CEOT lesions had calcification images, including 2 type â ¢ and 5 type â ¤ lesions. Six COC lesions had odontoma-like images. Moreover, 8 of 9 type â CEOTs were histologically Langerhans cell-rich subtype, which had a smaller size (with an average mesiodistal diameter of 17.8 mm) and were not associated with impacted teeth. CONCLUSION: COC lesions tended to originate from the anterior part of the jaw and exhibit cortex expansion, and were sometimes associated with odontoma. CEOT commonly occurred in the posterior jaw and had discontinued cortex. Two lesions had significantly different calcification map. Over 70% of COC lesions had calcification images, which were mostly scattered along one side of the cysts, far from the impacted teeth. Approximately 60% of CEOT lesions exhibited smaller size and non-calcification, and the remaining CEOT cases often had calcification images clustered around the impacted teeth.
Assuntos
Calcinose , Cisto Odontogênico Calcificante , Cistos Odontogênicos , Tumores Odontogênicos , Odontoma , Neoplasias Cutâneas , Dente Impactado , Masculino , Feminino , Humanos , Adulto , Cisto Odontogênico Calcificante/diagnóstico por imagem , Cisto Odontogênico Calcificante/patologia , Odontoma/patologia , Estudos Retrospectivos , Tumores Odontogênicos/diagnóstico por imagem , Cistos Odontogênicos/patologia , Calcinose/diagnóstico por imagemRESUMO
BACKGROUND: The objectives of the study are to employ modified Gallego staining in pathology and to specifically evaluate the hard tissue apposed to odontogenic epithelium in order to aid in the diagnosis. METHODS: Lillie's modification of Gallego's stain was utilized as a reference to create a fresh batch of the stain. The archival and running cases of 2021-2022 was screened for odontogenic pathologies, yielding approximately 46 cases, among these four cases were selected which required evaluation of the hard tissue matrix apposed to odontogenic epithelium for its characterization. The modified Gallego staining was employed in the soft tissue sections of these cases under controlled environment. The staining results were evaluated. RESULTS: The stain was used in cases to stain dentinoid deposition as green color in hybrid ameloblastoma, archegonous cystic odontoma, dentinogenic ghost cell tumor and also in other cases like calcifying odontogenic cyst. Bone presented as green in color, cells as pink color and collagen as green-pink. This helped in the correct diagnosis of these cases facilitating the correct treatment modality. CONCLUSIONS: In the field of oral pathology, there are a multitude of lesions which are odontogenic with the diagnosis of few of them hinging on the characterization of hard tissue matrix found in close approximation to odontogenic epithelium implying an inductive capability to the odontogenic epithelium. This particular modified Gallego stain has been able to help in diagnosis of few such cases in our repertoire of cases.
Assuntos
Ameloblastoma , Citrus , Cisto Odontogênico Calcificante , Tumores Odontogênicos , Odontoma , Tumores Odontogênicos/patologia , Ameloblastoma/patologia , Odontoma/patologia , Coloração e RotulagemRESUMO
BACKGROUND: CTNNB1 gene encodes beta catenin, a transcriptional activator of Wnt pathway involved in the pathogenesis of odontogenic lesions. Though located intramembranously, its translocation into cytoplasm and nucleus could trigger cell proliferation, inhibition of apoptosis, invasion and migration of the tumour cell. MATERIALS AND METHODS: Five electronic databases including MEDLINE by PubMed, Google scholar, Scopus, Trip, Cochrane library and EMBASE until 1 January 2023 without period restriction were thoroughly searched. Those articles that identified CTNNB1 mutation and beta catenin in odontogenic lesions were included for review. Risk of bias was analysed for each study using QUADAS 2 tool and Review Manager 5.3 was used to output its result. RESULTS: Thirty four published articles were included for data synthesis. A total of 1092 cases of odontogenic lesions were assessed for both CTNNB1 mutation and beta catenin expression. CTNNB1 mutation was observed in ameloblastoma, calcifying odontogenic cyst, calcifying cystic odontogenic tumour and all malignant odontogenic tumours. The beta catenin expression (nuclear and cytoplasmic) was maximum in odontogenic keratocyst and calcifying odontogenic cyst. The expression was variable in ameloblastomas, membranous in odontomas, calcifying cystic odontogenic tumour and nuclear in all malignant tumours. DISCUSSION AND CONCLUSION: High recurrence of odontogenic keratocyst and aggressiveness of solid ameloblastoma and malignant odontogenic tumours could be associated with the nuclear translocation of beta catenin. Disparity between CTNNB1 mutation and beta catenin expression within odontogenic lesions suggests alternate routes of beta catenin activation. The review results support the unique localisation of beta catenin as a helpful diagnostic factor in the pathogenesis of odontogenic lesions.
Assuntos
Ameloblastoma , Cisto Odontogênico Calcificante , Cistos Odontogênicos , Tumores Odontogênicos , Humanos , Ameloblastoma/genética , beta Catenina/genética , Tumores Odontogênicos/genética , Tumores Odontogênicos/patologiaRESUMO
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.
Assuntos
Ameloblastoma , Cisto Odontogênico Calcificante , Tumores Odontogênicos , Odontoma , Adulto , Criança , Humanos , Odontoma/complicações , Odontoma/diagnóstico por imagem , Odontoma/cirurgia , Tumores Odontogênicos/diagnóstico por imagem , Tumores Odontogênicos/cirurgia , Tumores Odontogênicos/patologia , Cisto Odontogênico Calcificante/diagnóstico por imagem , Cisto Odontogênico Calcificante/cirurgia , Cisto Odontogênico Calcificante/patologia , Ameloblastoma/diagnóstico por imagem , Ameloblastoma/cirurgia , Ameloblastoma/patologiaRESUMO
Introdução: Os cistos e tumores odontogênicos são lesões que apresentam comportamento biológico heterogêneo e patogênese ainda não totalmente esclarecida. A Yes-associated protein (YAP) atua como um regulador transcricional de genes envolvidos na proliferação celular e na apoptose, participando da ativação de vias associadas ao crescimento cístico e à progressão neoplásica. Objetivo: Analisar a expressão imuno-histoquímica da proteína YAP e correlacioná-la com marcadores envolvidos na proliferação celular e na apoptose em lesões odontogênicas epiteliais benignas. Metodologia: A amostra consistiu de 95 casos de lesões odontogênicas - 25 cistos dentígeros (CDs), 30 CO não sindrômicos (COs), 30 AMB convencionais (AMB-Cs) e 10 AMB unicísticos (AMB-Us) -, além de 10 espécimes de folículo dentários (FD). Foi realizada coleta dos dados clinico-demográficos dos casos, bem como análise morfológica para melhor caracterização da amostra. Os cortes histológicos foram submetidos à técnica imuno-histoquímica através da utilização dos anticorpos YAP, ciclina D1, Ki-67 e Bcl-2, e a análise da expressão destes foi realizada quali-quantitativamente, mediante metodologia adaptada. Os dados coletados seguiram para análise descritiva e estatística (p ≤ 0,05). Resultados: Houve discreta predileção por mulheres (n = 55; 57,6%) e por indivíduos na faixa etária dos 21 aos 40 anos (n = 50; 47,6%), sendo a região posterior de mandíbula mais afetada (64%). A análise da imunoexpressão de YAP revelou maiores níveis de expressão em COs, especialmente nas camadas basal e parabasal, seguido dos AMB-Us e AMB-Cs, que demonstraram moderada imunorreatividade, predominantemente nas células periféricas. Além disso, houve diferenças significativas quanto à imunoexpressão de YAP entre os grupos analisados, com existência de correlações positivas e estatisticamente significativas entre YAP e ciclina D1 em CDs e AMB-Us, e entre YAP e Ki-67 em AMB-Us (p < 0,05). Todavia, entre a imunoexpressão YAP e Bcl-2, foi verificada ausência de correlação estatisticamente significativa. Conclusões: A YAP pode exercer influência sobre a proliferação celular do epitélio de cistos e tumores odontogênicos, auxiliando, assim, na progressão das diferentes lesões odontogênicas (AU).
Background: Odontogenic cysts and tumors present heterogeneous biological behavior, and their etiopathogenesis is not fully understood yet. Yes-associated protein (YAP) acts as a transcriptional regulator of genes involved in cell proliferation and apoptosis, activating pathways associated with cystic growth and neoplastic progression. Objective: To analyze the immunohistochemical expression of YAP protein and correlate it with markers involved in cell proliferation and apoptosis in benign epithelial odontogenic lesions. Methods: The sample consisted of 95 cases of odontogenic lesions - 25 dentigerous cysts (DCs), 30 non-syndromic odontogenic keratocyst (OKCs), 30 conventional AMB (C-AMBs), and 10 unicystic AMB (UAMBs) -, in addition to 10 specimens of dental follicles (DF). Clinicodemographic data collection was carried out, as well as morphological analysis for better characterization of the sample. The histological sections were submitted to the immunohistochemical technique using YAP, cyclin D1, Ki-67, and Bcl-2 antibodies, and their immunoexpression analysis was performed qualitatively and quantitatively, through an adapted methodology. The collected data were submitted for descriptive and statistical analysis (p ≤ 0.05). Results: There was a slight predilection for women (n = 55; 57.6%) and individuals aged between 21 and 40 years (n = 50; 47.6%), with the posterior region of the mandible as the most affected site (64%). Analysis of YAP immunoexpression revealed higher expression levels in OKCs, especially in the basal and parabasal layers, followed by U-AMBs and C-AMBs, which showed moderate immunoreactivity, predominantly in peripheral cells. In addition, there were significant differences in YAP immunoexpression between the analyzed groups, with positive and statistically significant correlations between YAP and cyclin D1 in DCs and U-AMBs, and between YAP and Ki-67 in U-AMBs (p < 0.05). However, between YAP and Bcl-2 immunoexpression, there was no statistically significant correlation. Conclusions: YAP may influence on the cell proliferation of odontogenic cysts and tumors epithelium, thus helping with the progression of the different odontogenic lesions (AU) .
Assuntos
Proliferação de Células , Proteínas de Sinalização YAP/metabolismo , Proteínas com Motivo de Ligação a PDZ com Coativador Transcricional/metabolismo , Cisto Dentígero/patologia , Biomarcadores Tumorais , Prontuários Médicos , Estudos Retrospectivos , Interpretação Estatística de Dados , Apoptose , Cisto Odontogênico Calcificante/patologia , Estatísticas não Paramétricas , Proteínas Inibidoras de Diferenciação , Estudo Observacional , Achados Morfológicos e MicroscópicosRESUMO
Ghost Cell Odontogenic Carcinoma is a rare malignant odontogenic tumor that can appear as "de novo " or arises from malignant transformation of preexisting benign calcifying odontogenic cysts or dentinogenic ghost cell tumors after multiple recurrences. Ghost cell odontogenic carcinoma is histopathologically characterized by ameloblast-like islands of epithelial cells with aberrant keratinization, simulating a ghost cell, with varying amounts of dysplastic dentine. This article reports an extremely rare case of ghost cell odontogenic carcinoma with foci of sarcomatous change, involving maxilla andnasal cavity which arose from a previously existing recurrent calcifying odontogenic cysts in a 54-year-old man and reviews the features of this unusual and rare tumor. To the best of our knowledge, this is the first case of ghost cell odontogenic carcinoma with sarcomatous transformation to be reported till date. Owing to its rarity and unpredictability of clinical course, long -term follow up of patients with ghost cell odontogenic carcinoma, is mandatory for observation of recurrence and distant metastasis. Keywords: Ghost cell odontogenic carcinoma, maxilla, sarcoma, calcifying odontogenic cysts, ghost cells, odontogenic tumour.
Assuntos
Carcinoma , Neoplasias Maxilomandibulares , Cisto Odontogênico Calcificante , Tumores Odontogênicos , Sarcoma , Masculino , Humanos , Pessoa de Meia-Idade , Cisto Odontogênico Calcificante/patologia , Neoplasias Maxilomandibulares/patologia , Tumores Odontogênicos/patologiaRESUMO
BACKGROUND: Calcifying odontogenic cyst (COC) can be misdiagnosed as a lesion of endodontic origin when it is in close proximity to the periradicular tissue, and pulp sensibility tests are indispensable for differential diagnosis. However, when the adjacent teeth are necrotic or already endodontically treated, diagnosis becomes challenging. CASE DESCRIPTION: In this case report, a maxillary canine with an inadequate root canal treatment was considered as the source of an endodontic infection. Eight months after the retreatment, the patient sought treatment for a buccal intraoral swelling and a deep periodontal pocket and was referred for cone-beam computed tomography with a provisional diagnosis of a vertical root fracture. The tomography revealed an extensive lesion buccally to the roots of the canine and the adjacent vital lateral incisor. An unusual extended external resorption of the root of the vital lateral incisor was also evident. This finding shifted the diagnostic thinking toward a lesion of nonendodontic origin. The lesion was surgically enucleated, and the histopathologic examination confirmed the diagnosis of a COC. PRACTICAL IMPLICATIONS: Clinicians always must bear in mind the chance of a nonendodontic lesion masquerading as a lesion of endodontic origin. Cone-beam computed tomography should be considered in cases of doubt or in lesions refractory to endodontic treatment, as it can provide information on the clinicopathologic features of the lesion.
Assuntos
Cisto Odontogênico Calcificante , Humanos , Cisto Odontogênico Calcificante/patologia , Tomografia Computadorizada de Feixe Cônico/métodos , Dente Canino/diagnóstico por imagem , Dente Canino/cirurgia , Dente Canino/patologia , Incisivo/diagnóstico por imagem , Incisivo/cirurgiaRESUMO
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.
Assuntos
Ameloblastoma , Neoplasias Maxilomandibulares , Cisto Odontogênico Calcificante , Cistos Odontogênicos , Tumores Odontogênicos , Feminino , Humanos , Pessoa de Meia-Idade , Ameloblastoma/patologia , Tumores Odontogênicos/diagnóstico por imagem , Tumores Odontogênicos/cirurgia , Cisto Odontogênico Calcificante/diagnóstico por imagem , Cisto Odontogênico Calcificante/cirurgia , Cisto Odontogênico Calcificante/patologia , Neoplasias Maxilomandibulares/patologiaRESUMO
Odontogenic lesions are a heterogeneous group of diseases that presents differences in their biological behavior and the occurrence of variable inductive interactions. Calcifying odontogenic cyst (COC), or Gorlin's cyst, is a well-recognized example of these lesions. We describe a case of COC with AOT-like areas and highlights its morphological diversity. A 60-year-old pheoderma man presented with a large swelling in the anterior buccal region of the mandible. Panoramic radiography revealed a well-defined, unilocular, radiolucent lesion associated with important root resorption. Complete enucleation of the lesion was performed and the histopathological findings met the criteria for the diagnosis of COC, although the cyst exhibited unusual AOT-like features. The patient has been recurrence free for 6 months after surgery. COCs with AOT-like features are rare, and reflect the multipotentiality and complexity of the inductive effects of the odontogenic epithelium with the ectomesenchyme. Enucleation seems to be the most indicated treatment, similar to classical COC (AU)
As lesões odontogênicas são um grupo heterogêneo de patologias que apresentam diferenças no seu comportamento biológico, e ocorrência de interações indutivas variáveis. O cisto odontogênico calcificante (COC), ou cisto de Gorlin, é um exemplo bem conhecido destas lesões. Descrevemos um caso de COC com áreas adenomatóides e destacamos a sua diversidade morfológica. Paciente do sexo masculino, 60 anos de idade, apresentou um aumento de volume na região anterior da mandíbula. A radiografia panorâmica revelou uma lesão bem definida, unilocular e radiolúcida associada a uma reabsorção radicular importante. A enucleação completa da lesão foi realizada e os achados histopatológicos preencheram os critérios para o diagnóstico de COC, embora o cisto exibisse características adenomatóides pouco usuais. O paciente permanece livre de recidivas durante 6 meses após a cirurgia. Os COCs com características adenomatóides são raros, e refletem a multipotencialidade e complexidade dos efeitos indutivos do epitélio odontogênico com o ectomesênquima. A enucleação parece ser o tratamento mais indicado, semelhante ao COC clássico. (AU)
Assuntos
Humanos , Masculino , Pessoa de Meia-Idade , Patologia Bucal , Cirurgia Bucal , Cistos Odontogênicos , Cisto Odontogênico CalcificanteRESUMO
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.
Assuntos
Cisto Odontogênico Calcificante , Tumores Odontogênicos , Adulto , Biópsia , Humanos , Masculino , Cisto Odontogênico Calcificante/patologia , Cisto Odontogênico Calcificante/cirurgia , Tumores Odontogênicos/diagnóstico , Tumores Odontogênicos/patologia , Tumores Odontogênicos/cirurgiaRESUMO
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.
Assuntos
Doenças do Gato , Doenças do Cão , Cisto Odontogênico Calcificante , Tumores Odontogênicos , Animais , Doenças do Gato/diagnóstico por imagem , Doenças do Gato/cirurgia , Gatos , Doenças do Cão/diagnóstico por imagem , Doenças do Cão/cirurgia , Cães , Cisto Odontogênico Calcificante/diagnóstico , Cisto Odontogênico Calcificante/cirurgia , Cisto Odontogênico Calcificante/veterinária , Tumores Odontogênicos/diagnóstico por imagem , Tumores Odontogênicos/cirurgia , Tumores Odontogênicos/veterináriaRESUMO
Ghost cell odontogenic carcinoma (GCOC) is a rare malignant tumor of odontogenic origin, with only about 50 cases reported in the English literature so far. Histologically, it is characterized by ghost cells, dentinoid deposits, high grade malignant cellular features, and areas of necrosis and invasion. Having common histological features with other odontogenic ghost cell lesions (OGCL) like calcifying odontogenic cyst (COC) and dentinogenic ghost cell tumors, it is crucial to recognize GCOC malignant features, as it can be destructive and invasive, sometimes showing distant metastases and high recurrence rate. For this reason, it may entail more aggressive surgical approach and multimodal therapeutic regimen. Here we present a case report of GCOC arising in a previous COC, treated with surgical excision that showed persistence and recurrence after two years. The clinical and histological features of this rare occurrence are presented, in addition to the surgical approach, and a summary of literature review of OGCL.
Assuntos
Carcinoma , Neoplasias Maxilomandibulares , Cisto Odontogênico Calcificante , Cistos Odontogênicos , Tumores Odontogênicos , HumanosRESUMO
BACKGROUND: Ghost cells (GCs) are cells with distinct intracytoplasmic keratinization, which leads to the preservation of the cellular outline with a clear area corresponding to the previous nucleus location. GCs may show various patterns, such as degeneration, tissue granulation, and calcification. Their true nature and the mechanism regulating the conversion of odontogenic epithelial cells into GCs remain unclear. GC keratinization is different from normal keratinization as they are larger than keratotic squames, are frequently vacuolated, and have prominent nuclear membrane remnants. Few cystic lesions, odontogenic tumors, and non-odontogenic tumors, such as calcifying odontogenic cyst, craniopharyngioma, pilomatrixoma, odontoma, dentinogenic ghost cell tumor, and ghost cell odontogenic carcinoma, exhibit GCs as a typical feature. The Wnt and Notch signaling pathways play a role in the histogenesis of the neoplasms. HIGHLIGHT: The review clarifies the various proposed hypotheses of the histogenesis of GCs, including molecular pathogenesis. Diagnostic workup for the identification of GCs, including special staining and immunohistochemistry, has been extensively discussed. A stepwise algorithm for identifying odontogenic and non-odontogenic lesions containing GCs has been proposed. Additionally, the prognostic role of GCs in the lesions has been elucidated. CONCLUSION: Among the various hypotheses of the origin of GCs, we suggest that aberrant keratinization is the most accepted based on various immunohistochemical studies and special staining characteristics. GCs are a distinct characteristic entity of many odontogenic and non-odontogenic lesions; however, it remains controversial whether their presence has any pathognomonic role in the biological nature of these lesions.
Assuntos
Doenças do Cabelo , Neoplasias Maxilomandibulares , Cisto Odontogênico Calcificante , Tumores Odontogênicos , Neoplasias Hipofisárias , Neoplasias Cutâneas , Humanos , Neoplasias Maxilomandibulares/metabolismo , Cisto Odontogênico Calcificante/metabolismo , Tumores Odontogênicos/metabolismoRESUMO
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)
Assuntos
Humanos , Feminino , Adolescente , Cistos Odontogênicos , Cisto Odontogênico Calcificante/cirurgia , Faculdades de Odontologia , Biópsia/métodos , Diagnóstico por Imagem/métodos , Diagnóstico Clínico , Chile , Técnicas Histológicas , Cisto Odontogênico Calcificante/diagnóstico por imagem , Procedimentos Cirúrgicos Bucais/métodosRESUMO
OBJECTIVES: Calcifying odontogenic cysts (COC) and adenomatoid odontogenic tumors (AOT) have similar radiographic findings. We examined the radiographic and computed tomography (CT) images of patients histologically diagnosed with COC or AOT and identified their characteristic findings. METHODS: The subjects included 12 patients histologically diagnosed with COC or AOT (one female and five males per group), who underwent CT at our hospital between Nov 1998 and Jun 2019. The location of the lesion, impacted tooth, bone expansion, root resorption, tooth migration, calcified body, and presence or absence of a high-intensity zone in the marginal area of the lesion were examined. RESULTS: In patients with COC, five patients with COC exhibited bone expansion toward the buccal side. The lesion encompassing the crown was attached to the cement-enamel junction and contained a radiopaque lesion with a calcified body. In 6 patients with COC, irregularly shaped calcified bodies were observed with small tooth-like structures. In patients with AOT, all six patients with AOT exhibited bone expansion toward the buccal and lingual sides. The lesion encompasses a part of the tooth root or the entire tooth. Punctate calcification was observed within the lesion and the marginal area in three patients, and a high-intensity zone was observed in the marginal area of the lesion in two patients. CONCLUSION: We report imaging findings that may be characteristic of COC and AOT, suggesting that CT findings may be useful for differentiating between COC and AOT.
Assuntos
Cisto Odontogênico Calcificante , Cistos Odontogênicos , Tumores Odontogênicos , Ameloblastoma , Diagnóstico Diferencial , Feminino , Humanos , Masculino , Cisto Odontogênico Calcificante/diagnóstico por imagem , Cisto Odontogênico Calcificante/patologia , Tumores Odontogênicos/diagnóstico , Tumores Odontogênicos/patologia , Tomografia Computadorizada por Raios X/métodosRESUMO
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.
Assuntos
Cisto Odontogênico Calcificante , Cistos Odontogênicos , Odontoma , Adolescente , Feminino , Humanos , Masculino , Cisto Odontogênico Calcificante/complicações , Cisto Odontogênico Calcificante/diagnóstico , Cisto Odontogênico Calcificante/cirurgia , Odontoma/complicações , Odontoma/diagnóstico , Odontoma/cirurgiaRESUMO
BACKGROUND: Hybrid odontogenic lesions combine histopathological characteristics of two or more odontogenic cysts and/or tumours. The aim of this study was to evaluate the available data on hybrid odontogenic lesions (HOL) and to analyse their epidemiological/clinical features and biological behaviour. METHODS: An electronic search was done in January 2021 using multiple databases. Eligibility criteria encompassed publications with sufficient clinical and histological information to confirm the tumours' diagnoses. RESULTS: A total of 147 articles were included in this study, comprising 203 cases. Calcifying odontogenic cyst associated with odontoma (COC/OD) (37/18.2%) was the most common HOL. Females were more affected with a mean age of 24.9 years. Lesions presented as asymptomatic swellings, with a mean evolution time of 8.2 months (0.3-96), and mean tumour size of 4.8 cm (0.3-7). Radiographic aspects frequently showed radiolucent (139/68.4%) and unilocular (52/25.6%) images with well-defined limits (48/23.6%). The lesions mostly affected mandibular pre-molars (69/34%) and mandibular molars (69/34%) regions. Enucleation (89/43.8%) and surgical excision (59/29%) were the most common treatment modalities. The mean follow-up time was 33.8 months (0.5-216 months) and recurrences were observed in four cases (1.9%), all of which were central odontogenic fibroma associated with central giant cell granuloma (COF/CGCG). CONCLUSION: COC/OD is the most common HOL and recurrence is a rare event, being usually associated with the diagnosis of COF/CGCG.
Assuntos
Granuloma de Células Gigantes , Cisto Odontogênico Calcificante , Cistos Odontogênicos , Tumores Odontogênicos , Odontoma , Adulto , Feminino , Humanos , Cistos Odontogênicos/diagnóstico por imagem , Cistos Odontogênicos/epidemiologia , Tumores Odontogênicos/diagnóstico por imagem , Tumores Odontogênicos/epidemiologia , Odontoma/diagnóstico por imagem , Odontoma/epidemiologia , Adulto JovemRESUMO
The calcifying epithelial odontogenic tumor is a rare benign neoplasm that accounts for approximately 1% of all odontogenic tumors. Most of the cases occur in the posterior mandible, and a few involve the maxilla. Despite their relatively indolent biological behavior, tumors in the maxilla tend to grow fast. We report the case of a 33-year-old female patient exhibiting swelling in the right maxilla. An isodense area associated with an impacted supernumerary tooth was found on imaging examination. The histopathologic diagnosis was a calcifying epithelial odontogenic tumor. The treatment of choice was surgical removal of the lesion and associated dental elements. The patient has been followed up for 11 months and shows no signs of recurrence. Besides describing this case, we reviewed the literature on the association of calcifying epithelial odontogenic tumors with supernumerary teeth and found two case reports addressing this subject.