Maxillary dentigerous cyst showing squamous odontogenic tumor-like proliferation: surgical approach and literature review

Squamous odontogenic tumor (SOT) is a rare benign neoplasm of the jaw that likely arises from remnants of the dental lamina. It is a slow-growing lesion, with a radiolucent appearance in the central variant. Microscopically, SOT shows islands of squamous epithelium supported by fibrous stroma. In rare cases, squamous odontogenic tumor-like proliferation (SOT-LP) can be observed arising from odontogenic cysts (SOT-LPOC). Herein, we describe the case of a 42-year-old man who presented with discreet bleeding in the maxillary gingiva. Imaging revealed a well-defined, ovoid-shaped lesion with sclerotic margins involving tooth #18 in the intraosseous location. Fine needle aspiration supported the cystic nature of the lesion. After surgery, microscopy revealed a dentigerous cyst showing SOT-LP features. There was no recurrence after a 3-year follow-up. To the best of our knowledge, this is the first report of a dentigerous cyst showing SOT-LP features in the maxilla. Such cysts should be identified to avoid misdiagnosis, with the finding having therapeutic and prognostic implications.

Colgajo temporal en la reconstrucción de un defecto maxilar por exéresis de carcinoma ameloblástico / Temporalis muscle flap in the reconstruction of a maxillary defect caused by exeresis of ameloblastic carcinoma

RESUMEN Introducción: El carcinoma ameloblástico es una entidad rara que surge como una neoplasia primaria o a partir de un ameloblastoma preexistente. El colgajo de músculo temporal es una opción terapéutica frecuentemente empleada para la reconstrucción del defecto resultante luego de la exéresis quirúrgica. Objetivo: Presentar un caso clínico de restauración estética y funcional mediante reconstrucción con colgajo temporal de un defecto maxilar por exéresis de carcinoma ameloblástico, dada la infrecuente presentación de esta entidad. Caso clínico: Mujer de 49 años de edad, que refiere "una bola" en el paladar de 9 meses de evolución. Al examen físico facial presenta aumento de volumen en región infraorbitaria izquierda. Se realizó una tomografía axial computarizada en la que se constató la presencia de imagen hiperdensa en seno maxilar izquierdo con calcificación en su interior, produciendo lisis del hueso nasal y hueso cigomático infiltrando partes blandas. Se tomó muestra para biopsia que informó tumor de alto grado de malignidad correspondiente a carcinoma ameloblástico. En estudio radiográfico de tórax no se apreció presencia de metástasis pulmonar. Se realizó maxilarectomía de infra y mesoestructura, resección de la lesión con margen oncológico de seguridad y se reconstruyó el defecto palatino con colgajo pediculado del músculo temporal. Se indicó quimio y radioterapia como terapia adyuvante al tratamiento quirúrgico. Se mantuvo el chequeo posoperatorio mostrándose buena evolución clínica y una epitelización secundaria del músculo temporal en el área palatina con restauración de las funciones. Conclusiones: Se presentó un caso clínico de carcinoma ameloblástico, entidad patológica de escasa frecuencia. La cirugía constituyó el pilar de tratamiento utilizado. Una vez realizada la resección quirúrgica se reconstruyó el defecto palatino, utilizándose el colgajo del músculo temporal, opción útil para lograr el restablecimiento de las funciones estéticas y funcionales como la deglución y fonación(AU)

ABSTRACT Introduction: Ameloblastic carcinoma is a rare condition emerging as a primary neoplasm or from a preexisting ameloblastoma. Temporalis muscle flap is a therapeutic option frequently used for reconstruction of the defect resulting from surgical exeresis. Objective: Present a clinical case of esthetic and functional restoration by reconstruction with temporalis muscle flap of a maxillary defect caused by exeresis of an ameloblastic carcinoma. The case is presented because of the infrequent occurrence of this condition. Clinical case: A female 49-year-old patient reports "a lump" in her palate of nine months evolution. Physical examination finds an increase in volume in the left infraorbital region. Computed axial tomography was indicated, which revealed the presence of a hyperdense image in the left maxillary sinus with internal calcification causing lysis of the nasal bone and the zygomatic bone, and infiltrating soft tissue. A sample was taken for biopsy, which reported a tumor with a high degree of malignancy corresponding to ameloblastic carcinoma. Chest radiography did not show the presence of lung metastasis. Infra- and mesostructure maxillectomy was performed, the lesion was removed with a surgical safety margin, and the palatine defect was reconstructed with a pediculated temporalis muscle flap. Chemo- and radiotherapy were indicated as adjuvants to the surgical treatment. Postoperative follow-up found good clinical evolution and secondary epithelization of the temporalis muscle in the palatine area with restoration of functions. Conclusions: A clinical case was presented of ameloblastic carcinoma, a condition with a low frequency of occurrence. Surgery was the basic component of the treatment applied. Once surgical resection was performed, the palatine defect was reconstructed by means of a temporalis muscle flap, a useful option to achieve the restoration of esthetic and biological functions, such as swallowing and speech(AU)

Immunoglobulin G4-related disease of the maxillofacial region: a rare case / Enfermedad relacionada con inmunoglobulina G4 en el área maxilofacial: un caso raro

IgG4-related disease is a systemic, multifocal, immune-mediated disorder that can affect multiple organs and may present as a tumor, with rare cases described in the maxillofacial region. A female patient, 53 years old, presenting tumor-like mass in the right mandibular region. Magnetic resonance imaging suggested well circumscribed nodular lesion adjacent to the branch / body of the mandible, extending posteriorly to the masseter muscle. During the surgical procedure of excision, a lesion was observed adhering to the right masseter muscle, but it was possible to remove it completely. Histopathological and immunehistochemical analysis suggested diagnosis of IgG4-related disease, furthermore, IgG4 serum count was increased. Actually, the patient continues on periodical followups in our service and by other specialties. Can be concluded that precise diagnosis of this pathology depends on many factors, being challenging and the treatment involves multidisciplinary evaluation due to the possibility of involvement of several other organs.

La enfermedad relacionada con IgG4 es una condición sistémica, multifocal, mediada por una alteración de la respuesta inmune que puede afectar diferentes órganos o puede presentarse como un tumor, raramente descrito en el área maxilofacial. Se describe el caso de una paciente de sexo femenino de 53 años de edad, presentando una masa tumoral en el ángulo mandibular derecho. La resonancia magnética sugirió un área nodular bien delimitada adyacente al cuerpo mandibular y extendida posteriormente hasta el musculo masetero. Durante la escisión quirúrgica, la lesión se presentaba adherida al musculo de forma lateral siendo posible el retiro total de la lesión. El estudio histopatológico e inmunohistoquimico determinó el diagnóstico de enfermedad relacionada con IgG4 presentando un conteo de igG4 aumentado. Actualmente, la paciente continua con seguimiento por la especialidad. Se puede concluir que el diagnóstico preciso de esta patología depende de algunos factores; el tratamiento debe ser multidsciplinario debido a la inclusión de diferentes órganos en la enfermedad.

Ameloblastoma with distinctive granular cell pattern: an 8 case study

Ameloblastoma with granular cell pattern (AGC) is classified as an unusual histological subtype of solid/multicystic ameloblastoma, characterized by granular changes in stellate-like cells located within the inner portion of the epithelial follicles. Studies have revealed that lysosomal overload causes cytoplasmic granularity; however, the mechanism involved remains poorly understood. Here we report on eight cases of granular cell ameloblastoma, in the posterior region of the mandible. The age of the patients included in this case series ranged from 35 -64 years old and 87.5% of cases occurred in non-Caucasians, with a slight gender predilection for men (62.5%). There was no evidence of recurrence, and the majority of the cases were treated with surgical resection (87.5%). All tumors displayed histopathological features consistent with the diagnosis of ameloblastoma with granular cell pattern.

Expresión inmunohistoquímica de ki-67 y p53 en tumor odontogénico queratoquístico en comparación con ameloblastoma multiquístico / Immunohistochemical expression of Ki-67 and p53 in odontogenic keratocysts in comparison with ameloblastomas

Introducción: Ha sido descrita la inmunodetección de p53 y Ki-67 en el tumor odontogénico queratoquístico (TOQ) y en ameloblastomas mutiquísticos (AM). Sin embargo, hay escasa y contradictoria evidencia respecto de la comparación de estos dos marcadores entre estas neoplasias. Su estudio podría contribuir a comprender las diferencias que presentan en su comportamiento clínico y ser un complemento discriminatorio al momento de definir tratamiento, pronóstico y recidiva. Objetivo: Comparar el recuento de inmunomarcación de p53 y Ki-67 en células epiteliales de TOQ y AM, presentes en el registro de biopsias del Instituto de Referencia de Patología Oral (IREPO), de la Facultad de Odontología de la Universidad de Chile entre los años 2000-2011. Metodología: Estudio observacional de corte transversal. Se estudiaron mediante inmunohistoquímica 8 casos de TOQ y 6 casos de AM con diagnóstico histopatológico según la clasificación de la OMS del 2005. Las muestras se encontraban en bloques de parafina, fijadas en formalina. Resultados: Los datos mostraron una distribución normal en cuanto al número de células positivas para ambos inmunomarcadores. No hubo diferencias estadísticamente significativas en la inmunomarcación de Ki-67 y p53 para TOQ y AM. Sin embargo, en ambas neoplasias, la inmunomarcación de p53 fue mayor respecto a Ki-67, con una diferencia estadísticamente significativa tanto en TOQ (p=0.0134) como en AM (p=0.0079). Conclusión: Los resultados sugieren que la inhibición de apoptosis predominó en ambos tumores por sobre la multiplicación celular. Estas diferencias podrían relacionarse con su potencial de crecimiento.

Introduction: p53 and Ki-67 immunodetection have been described in the keratocystic odontogenic tumor (TOQ) and multicystic ameloblastomas (AM). However, there is limited and contradictory evidence regarding the comparison of these two markers between these neoplasias. Their study could help to understand the differences that occur in their clinical behavior and be a complement when defining discriminatory treatment, prognosis and recurrence. Objective: To compare the immunomarking count of p53 and Ki-67 in epithelial cells in AM and TOQ present in the biopsies registered at the Oral Pathology Reference Institute (IREPO), Faculty of Dentistry, University of Chile from 2000 to 2011. Methods: Cross-sectional observational study. 8 cases of TOQ and 6 cases of AM with histopathological diagnosis according to the WHO classification of 2005 were studied using immunohistochemistry. The samples were formalin-fixed and paraffin-embedded. Results: The data showed a normal distribution in the number of positive cells for both immunomarkers. There were no statistically significant differences in the Immunohistochemical expression of Ki-67 and p53 of TOQ and AM. However in both tumors, the immunohistochemical expression of p53 was higher compared to Ki-67, with a statistically significant difference in TOQ (p=0.0134) and AM (p=0.0079). Conclusion: The results suggest that inhibition of apoptosis in both tumors predominated over cell multiplication. These differences may be related to their growth potential.

Odontogenic tumors: a 14-year retrospective study in Santa Catarina, Brazil

Odontogenic tumors (OTs) are lesions that develop exclusively on maxillary bones, and form a heterogeneous group. They vary from hamartomatous lesions to benign and malign tumors. Although they are rarely observed in dentistry clinics, it is extremely important for the dentist to be aware of them. The aim of this study was to investigate the incidence of odontogenic tumors diagnosed in the population of Florianópolis, Santa Catarina, Brazil. Cases of odontogenic tumors were selected from the anatomopathological diagnostic services at Federal University of Santa Catarina from 1998 to 2011. Clinical data on these cases were collected from biopsy reports and patient files. Seventy-eight cases of odontogenic tumors were surveyed. Of these diagnoses, 51% were keratocystic odontogenic tumors (KCOTs); the remaining cases were mainly ameloblastomas and odontomas. The most frequently observed lesion in this retrospective study was KCOT (more than half of cases). Thus, this study shows that modifying the classification of the OTs altered the frequency of the lesions, possibly making KCOT the most common lesion observed in diagnostic services worldwide.

Ossifying Fibroma of the Jaws: A Clinicopathological Case Series Study

The aim of this study was to assess the clinical, radiographic and microscopic features of a case series of ossifying fibroma (OF) of the jaws. For the study, all cases with OF diagnosis from the files of the Oral Pathology Laboratory, University of Ribeirão Preto, Ribeirão Preto, SP, Brazil, were reviewed. Clinical data were obtained from the patient files and the radiographic features were evaluated in each case. All cases were reviewed microscopically to confirm the diagnosis. Eight cases were identified, 5 in females and 3 in males. The mean age of the patients was 33.7 years and most lesions (7 cases) occurred in the mandible. Radiographically, all lesions appeared as unilocular images and most of them (5 cases) were of mixed type. The mean size of the tumor was 3.1 cm and 3 cases caused displacement of the involved teeth. Microscopically, all cases showed several bone-like mineralized areas, immersed in the cellular connective tissue. From the 8 cases, 5 underwent surgical excision and 1 patient refused treatment. In the remaining 2 cases, this information was not available. In conclusion, OF occurs more commonly in women in the fourth decade of life, frequently as a mixed radiographic image in the mandible. Coherent differential diagnoses are important to guide the most adequate clinical approach. A correlation between clinical, imaginological and histopathological features is the key to establish the correct diagnosis.

O objetivo deste estudo foi analisar as características clínico-radiográficas e microscópicas de uma série de casos de fibroma ossificante (FO). Para o estudo, todos os casos com diagnóstico de FO do arquivo do Laboratório de Patologia Bucal da Universidade de Ribeirão Preto, Ribeirão Preto, São Paulo, Brasil, foram estudados. Os dados clínicos foram coletados das fichas de encaminhamento das lesões ao Laboratório. As características radiográficas foram avaliadas em cada caso. Todos os casos foram revisados microscopicamente para confirmação do diagnóstico. Oito casos foram identificados, 5 em mulheres e 3 em homens. A idade média dos pacientes foi de 33,7 anos e a maioria das lesões (7 casos) ocorreu na mandíbula. Radiograficamente todas as lesões se apresentavam como imagens uniloculares e a maioria (5 casos) se mostravam como imagem mista. O tamanho médio do tumor foi de 3,1 cm e 3 casos causavam deslocamento dos dentes envolvidos. Microscopicamente, todos os casos apresentavam várias áreas mineralizadas semelhantes a osso, imersas em um tecido conjuntivo celularizado. Dos 8 casos, 5 foram submetidos a excisão cirúrgica da lesão e 1 paciente recusou tratamento. Nos outros 2 casos, essa informação não estava disponível. Conclui-se que o FOs ocorrem mais comumente na mandíbula de pacientes do gênero feminino durante a quarta década de vida e se apresentam frequentemente como uma imagem radiográfica mista. Diagnósticos diferenciais coerentes são importantes para guiar a conduta clínica mais adequada. A correlação entre as características clínicas, imaginológicas e histopatológicas é a chave para estabelecer o diagnóstico correto.

Carcinoma primario intraóseo / Primary intra-osseous carcinoma

El carcinoma primario intraóseo (PIOC) es un tumor poco frecuente, definido como carcinoma escamoso que se desarrolla en huesos maxilares, no teniendo conexión inicial con mucosa ni piel adyacente. Es localmente agresivo, con una incidencia de metástasis en ganglios regionales del 28% y en pulmón del 5%, en el momento del diagnóstico. Su origen puede ser de novo o a partir de otros tumores odontogénicos. Los huesos maxilares son los únicos que tienen en su interior tejidos epiteliales, por lo cual esta neoplasia se localiza exclusivamente en este sitio, predominantemente en la mandíbula. Los criterios diagnósticos del PIOC incluyen: histopatología de carcinoma escamocelular, ausencia de compromiso de mucosa oral y senos paranasales, descartando metástasis de un sitio distante en base a estudios clínicos y métodos complementarios. El tratamiento de elección consiste, siempre que sea posible, en la exéresis con criterios oncológicos, y radio y/o quimioterapia adicional. Se requiere además, cirugía reconstructiva con injerto y/o prótesis con fines estéticos y funcionales. Presentamos el caso de un varón de 72 años, que consultó por molestias en maxilar inferior tres meses después de la extracción de un molar. Se efectuó biopsia por curetaje y luego se resecó el maxilar inferior con vaciamiento ganglionar. El estudio histopatológico mostró un carcinoma escamoso pobremente diferenciado, infiltrante en hueso maxilar, con hallazgos morfológicos que lo vinculaban a quiste odontogénico residual, y metástasis en 15 de 48 ganglios aislados. Se realizó radioterapia postquirúrgica, falleciendo a los 30 meses del diagnóstico por deterioro progresivo.

Primary intra-osseous carcinoma (PIOC) is a rare tumor, defined as squamous cell carcinoma that develops in the jaw bones, having no initial connection to adjacent skin or mucosa. It is locally aggressive, with metastases to regional lymph nodes, (28% of cases) and lung (5% of cases) at the time of diagnosis. Its origin may be di novo or from other odontogenic tumors. The maxillary bones have epithelial tissues; therefore this neoplasm is located exclusively on this site, predominantly in the jaw. PIOC diagnostic criteria are strict and include: squamous cell carcinoma histopathology, lack of commitment and sinus mucosa, ruling out the possibility of metastasis from a distant site with a thorough clinical study and complementary methods. The treatment is, whenever possible, oncologic resection, additional radio and / or chemotherapy. Reconstructive surgery with graft and / or prostheses for aesthetic and functional are also required. We report the case of a 72 years old man who consulted for sore jaw three months after molar extraction. Curettage biopsy was performed and then resected mandible with lymphadenectomy. Histopathological examination showed a poorly differentiated squamous cell carcinoma, infiltrating jawbone with morphological findings linking him to residual odontogenic cyst and metastatic lymph nodes in 15 of 48 isolates. Postoperative radiotherapy was performed, he died at 30 months of diagnosis by progressive deterioration.

Lesiones proliferativas más frecuentes del complejo bucomaxilofacial / Most frequent proliferative lesions of the Buco Maxillofacial Complex

Introducción: las lesiones proliferativas son entidades que se presentan en la cavidad bucal. Algunas son de origen traumático y otras son neoplasias. Con frecuencia no son bien diagnosticadas en la clínica, por la similitud que pueden presentarse entre ellas. Objetivo: determinar las características de las lesiones proliferativas diagnosticadas y la coincidencia entre el diagnóstico clínico y el histopatológico. Método: se realizó un estudio retrospectivo de las biopsias procesadas en cinco años en el Departamento de Patología de la Facultad de Estomatología de La Habana atendiendo a edad y sexo del paciente, tipo de lesión, localización anatómica y coincidencia entre el diagnóstico clínico y el histopatológico. Resultados: se encontró que de las lesiones proliferativas, las pseudotumorales aportaron el 18,1 por ciento y las neoplasias benignas el 5,8 por ciento. En el grupo de edad de 10 a 19 años las lesiones pseudotumorales representaron el 7,78 por ciento y las neoplasias benignas el 1,78 por ciento. En el grupo de 60 años o más, las lesiones pseudotumorales aportaron el 22,67 por ciento del total de lesiones proliferativas. El sexo femenino resultó afectado por lesiones pseudotumorales en 61,3 por ciento y las neoplasias benignas afectaron al sexo masculino en un 55 por ciento. La hiperplasia fibroepitelial representó el 54,22 por ciento de las pseudotumorales, mientras que el nevus aportó el 8,67 por ciento entre las neoplasias benignas. Los diagnósticos clínicos de las lesiones pseudotumorales coincidieron en un 50,4 por ciento con los histológicos, para las neoplasias benignas la coincidencia fue del 53,2 por ciento. Conclusiones: las lesiones pseudotumorales predominaron sobre las neoplasias benignas, con un incremento marcado de ambos tipos de lesiones a partir de los 40 años. En el sexo femenino predominaron las lesiones pseudotumorales, y en el masculino predominaron las neoplasias benignas. Se encontró una baja coincidencia entre el diagnóstico clínico e histopatológico para ambos tipos de lesiones(AU)

Introduction: proliferative lesions are entities that are present in the buccal cavity. Some of them are of traumatic origin and others are neoplasias. Frequently, they are not well diagnosed in the clinical practice because of the similarity that can exist among them. Objective: to determine the characteristics of the proliferative lesions diagnosed and the coincidences between clinical and histopathological diagnosis Method: a retrospective study of the biopsies processed during five years in the Department of Oral Pathology of the Faculty of Stomatology of Havana was conducted dealing with age, sex, type of lesion, anatomic location and similarities between the clinical and histopathological diagnosis. Results: it was found that, of the proliferative lesions, the pseudotumoral ones reported the 18.1 percent and the benign neoplasias, the 5.8 percent. In the age group 10-19 years, pseudotumoral lesions represented the 7.78 percent and the benign neoplasias the 1,78 percent. In the group of 60 years or older, pseudotumoral lesions reported the 22.67 percent of the total of proliferative lesions. The female sex resulted to be affected by pseudotumoral lesions in a 61,3 percent and the benign neoplasias effected the male sex in a 55 percent. Fibroepithelial hyperplasia represented the 54.22 percent of the pseudotumoral ones, whereas the nevus reported the 8.67 percent among the benign neoplasias. The clinical diagnosis of the pseudotumoral lesions matched up a 50.4 percent with the histological; for benign neoplasias, the coincidence was 53.2 percent. Conclusions: pseudotumoural lesions predominated on benign neoplasias, with a marked increase in both types of lesions after 40 years. Pseudotumoural lesions predominated in the female sex, whereas the benign neoplasias predominated in the male sex. There was a low coincidence between the clinical and histopathological diagnosis for both types of lesions(AU)

Carcinoma ameloblástico de mandíbula / Carcinoma ameloblástico de la mandíbula / Ameloblastic carcinoma of the mandible

O termo carcinoma ameloblástico é usado para descrever ameloblastomas com características histológicas de malignidade. Lesões desse tipo podem ocorrer em uma extensa faixa etária, sendo mais comum na quarta década de vida. Não há predileção aparente por sexo, a área mais comumente afetada é a porção posterior da mandíbula, envolvendo em menor proporção a maxila. Por se tratar de um tumor com poucos casos descritos na literatura, o objetivo deste trabalho foi relatar um caso de carcinoma ameloblástico em paciente do sexo feminino, 38 anos, com queixa de dor e assimetria facial. Ao exame clínico intrabucal observou-se um aumento de volume em região posterior de mandíbula, do lado direito. A tomografia computadorizada revelou adelgaçamento das corticais com presença de área hipodensa homogênea. Foi realizada biópsia incisional cujo diagnóstico histológico inicial foi de ameloblastoma, sem características citológicas de malignidade. Baseando-se no exame histopatológico, optou-se pelo tratamento cirúrgico da lesão através da ressecção parcial da mandíbula. A peça cirúrgica foi encaminhada ao laboratório obtendo-se novo diagnóstico de carcinoma ameloblástico. Provavelmente, a diferença do diagnóstico histológico final para o inicial pode estar relacionada ao fato de o carcinoma ameloblástico apresentar áreas focais semelhantes ao ameloblastoma. Após 6 meses da cirurgia, a paciente apresentou metástase tumoral no pulmão e encontra-se sob supervisão médica(AU)

El término carcinoma ameloblástico es utilizado para describir ameloblastomas con características histológicas de malignidad. Este tipo de lesión puede ocurrir en cualquier edad, pero se puede observar con mayor frecuencia en la cuarta década de vida. No existe aparentemente un sexo predominante. El área más comúnmente afectada es la porción posterior de la mandíbula y afecta en menor proporción el maxilar. Por ser un tumor con pocos casos relatados en la literatura, el objetivo de este trabajo fue presentar un caso de carcinoma ameloblástico en un paciente del sexo femenino con 38 años de edad, que acudió a la consulta por presentar síntomas dolorosos y asimetría facial. Al examen clínico intrabucal se observó un aumento de volumen en la región posterior derecha de la mandíbula. La tomografía computadorizada mostró un adelgazamiento de las corticales con presencia de un área hipodensa homogénea. Fue realizada una biopsia incisional y su diagnóstico histológico inicial fue de un ameloblastoma sin características citológicas de malignidad. Después de realizado el examen histopatológico se optó por el tratamiento quirúrgico de la lesión con resección parcial de la mandíbula. La estructura quirúrgica fue enviada al laboratorio, donde se obtuvo un nuevo diagnóstico de carcinoma ameloblástico. Probablemente la diferencia del diagnóstico histológico final con el inicial pudo estar relacionada con el hecho de que el carcinoma ameloblástico presenta áreas en foco semejantes al ameloblastoma. Luego de 6 meses del procedimiento quirúrgico, la paciente presentó metástasis tumoral en el pulmón y se encuentra bajo supervisión médica(AU)

The term ameloblastic carcinoma is used to describe the ameloblastomas with histological characteristics of malignancy. This type of lesion may to occur in any age, but it may be more frequent observed in the fourth decade of life. The is not apparently a predominance sexual. The commonest involved area is the posterior portion of mandible involving in less extent the maxillary. This is a tumor with few cases mentioned in the literature, thus, the aim of present paper was to present a case of ameloblastic carcinoma in a female patient aged 38 came to consultation due to painful symptoms and facial asymmetry. In intrabuccal clinical examination it was noted a volume increase in the right posterior region of mandible. The computed tomography showed a slimming of the cortex with presence of a homogenous hypo-dense area. An incisional biopsy was made and its initial histological diagnosis was an ameloblastoma without histological characteristics of malignancy. After histopathological examination the surgical treatment of lesion was prescribed with a partial resection of mandible. The surgical structure (sample) was sent to laboratory to obtain a new diagnosis of ameloblastic carcinoma. It is probable that the difference of final histological diagnosis with the initial one could be related to the fact that the ameloblastic carcinoma has focal areas similar to ameloblastoma. After 6 months of surgical procedure the patient had tumoral lung metastasis and is under medical supervision(AU)

Estudo demográfico, clínico e terapêutico do ameloblastoma: análise de 40 casos / Ameloblastoma demographic, clinical and treatment study: analysis of 40 cases

As lesões odontogênicas representam cerca de 1% dos tumores da cavidade bucal, sendo o ameloblastoma a afecção mais frequente. Trata-se de um tumor de origem epitelial, que afeta principalmente a mandíbula e, raramente, a maxila. Clinicamente, apresenta-se como tumor de crescimento lento, assintomático. Apesar de ser um tumor benigno, cursa com comportamento invasivo com alta taxa de recidiva, se não tratado adequadamente. OBJETIVO: Descrever os casos de ameloblastoma em serviço de referência. MÉTODOS: Análise retrospectiva de 40 casos. As variáveis analisadas foram: idade, gênero, etnia, localização do tumor, tipo de tratamento, complicação e recorrência. RESULTADOS: O gênero mais atingido foi o masculino, 21 casos (52,5%), com predomínio da etnia caucasiana - 24 casos (60%). A média de idade foi de 35,45 anos, a localização mais comum foi na mandíbula - 37 casos (92,5%). Assimetria facial a queixa mais frequente. Dos 40 casos, 33 foram submetidos a procedimento cirúrgico. Dos que tiveram tratamento cirúrgico, 24 (72,72%) foram submetidos à ressecção segmentar, com recidiva em quatro (12,12%) casos. CONCLUSÃO:O ameloblastoma apresenta possibilidade de recidiva quando o tratamento cirúrgico não é realizado com ressecção ampla da lesão com margens de segurança.

Dental lesions represent about 1% of oral cavity tumors being ameloblastoma the most common one. It is a tumor of epithelial origin that mainly affects the jaw, and less commonly the maxilla. Its clinical presentation is that of an asymptomatic slow-growing tumor. Despite being a benign tumor, it has an invasive behavior with a high rate of recurrence if not treated properly. OBJECTIVE: To describe the cases of ameloblastoma in a reference department. METHODS: Retrospective analysis of 40 cases. The variables analyzed were: age, gender, ethnicity, tumor location, type of treatment, complications and recurrence. RESULTS: The most affected gender was male - 21 cases (52.5%); with a predominance of Caucasians - 24 cases (60%). The mean age was 35.45 years; the most common location was in the jaw - 37 cases (92.5%). Facial asymmetry was the most frequent complaint. Of the 40 cases, 33 were submitted to surgery. Of those submitted to surgery, 24 (72.72%) underwent segmental resection, with recurrence in 4 (12.12%) cases. CONCLUSION:Ameloblastoma may relapse when treatment is not performed with broad surgical resection of the lesion with wide safety margins.

Cell proliferation and apoptosis in ameloblastomas and keratocystic odontogenic tumors

A high proliferative activity of the odontogenic epithelium in ameloblastoma (AM) and keratocystic odontogenic tumor (KOT) has been demonstrated. However, no previous study has simultaneously evaluated cell proliferation and apoptotic indexes in AM and KOT, comparing both lesions. The aim of this study was to assess and compare cell proliferation and apoptotic rates between these two tumors. Specimens of 11 solid AM and 11 sporadic KOT were evaluated. The proliferation index (PI) was assessed by immunohistochemical detection of Ki-67 and the apoptotic index (AI) by methyl green-pyronine and in situ DNA nick end-labelling methods. KOT presented a higher PI than AM (p<0.05). No statistically significant difference was found in the AI between AM and KOT. PI and AI were higher in the peripheral cells of AM and respectively in the suprabasal and superficial layers of KOT. In conclusion, KOT showed a higher cell proliferation than AM and the AI was similar between these tumors. These findings reinforce the classification of KOT as an odontogenic tumor and should contribute to its aggressive clinical behavior.

Uma elevada atividade proliferativa do epitélio odontogênico em ameloblastoma (AM) e tumor odontogênico ceratocístico (TOC) tem sido demonstrada. Entretanto, não há estudos prévios avaliando simultaneamente os índices de proliferação celular e apoptótico em AM e TOC, comparando ambas as lesões. O objetivo desse estudo foi avaliar e comparar os índices de proliferação celular e apoptótico entre esses dois tumores. Onze amostras deAM sólido e 11 amostras de TOC esporádico foram avaliadas. O índice de proliferação celular foi avaliado por meio da imunomarcação para o antígeno Ki-67 e o índice apoptótico pelas técnicas demetyl-green-pironina e TUNEL. O TOC apresentou um índice de proliferação celular maior que o AM (p<0,05). Nenhuma diferença estatística foi encontrada no índice apoptótico entre AM e TOC. Os índices de proliferação celular e apoptótico foram maiores nas células da camada periférica do AM e, respectivamente, nas camadas suprabasal e superficial do TOC. Em conclusão, o TOC apresentou proliferação celular maior que o AM e o índice apoptótico foi similar entre estes tumores. Estes achados reforçam a classificação do TOC como um tumor odontogênico e podem contribuir para o seu comportamento clínico agressivo.

Mandibular ameloblastoma in a 10-year-old child: case report and review of the literature / Ameloblastoma mandibular en niño de 10 años: reporte de un caso y revisión de la literatura

The ameloblastoma according to the classification of odontogenic tumors by WHO in 2005, is classified as a benign neoplasm of odontogenic epithelial origin. One to three percent of tumors and cysts of the jaws are comprised of ameloblastomas. The tumor is locally aggressive, but often asymptomatic, showing a slow growth which is manifested as a facial swelling or radiographic incidental finding. On clinical examination, the tumor can cause symptoms such as pain, ulceration, tooth mobility, root resorption and malocclusion. Ameloblastomas have a high rate of recurrence if not completely removed. It occurs in almost all age groups, but is mainly diagnosed in the third or fourth decade of life. The tumor is very rare in children. We present an unusual case of a solid/multicystic ameloblastoma of the mandible in a 10-year-old girl. In addition, a brief review of the literature on reported cases of this pathology in children is also presented...

El ameloblastoma según la clasificación de tumores odontogénicos de la OMS del 2005 lo clasifica como una neoplasia benigna de origen epitelial odontogénico. Compromete el 1-3% de neoplasias y quistes maxilares. El tumor es agresivo localmente, pero muchas veces asintomático; presenta un lento crecimiento que se manifiesta como un aumento de volumen facial o un hallazgo incidental radiográfico. Al examen clínico el tumor puede causar síntomas como dolor, ulceración, reabsorción radicular con movilidad dentaria y maloclusión. El ameloblastoma posee gran tasa de recurrencia si no es totalmente removido. Se presenta en casi todos los grupos etarios pero principalmente se diagnostica en la tercera o cuarta década de vida, el tumor es muy poco común en niños. El tratamiento del ameloblastoma es controversial y debido a la distinta incidencia y comportamiento en niños, hace las consideraciones quirúrgicas diferentes a los adultos. Por lo que presentamos un inusual caso de un ameloblastoma solido/multiquístico mandibular en una niña de 10 años. Además de una breve revisión de la literatura sobre casos reportados de esta patología en niños...

Ameloblastoma in Jamaica - predominantly unicystic: analysis of 47 patients over a 16-year period and a case report on re-entry cryosurgery as a new modality of treatment for the prevention of recurrence / El ameloblastoma en Jamaica - predominantemente unicístico: análisis de 47 pacientes por un período de más de 16 años y un reporte de caso de criocirugía de reentrada como nueva modalidad de tratamiento para prevenir la recurrencia

OBJECTIVE: To analyse all the cases of ameloblastoma seen in the two major public Hospitals in Jamaica over a 16year period, and to compare the results with what has been previously documented by other authors in the literature. A new treatment modality for prevention of recurrence of ameloblastoma is described. SUBJECTS AND METHODS: The case files of patients histologically diagnosed to have ameloblastoma at both the Kingston Public Hospital and Cornwall Regional Hospital in Jamaica from 1980 to 1995 were retrieved and information about this odontogenic tumour was documented. The analysis revealed that uncystic ameloblastoma was predominant (95%) and solid ameloblastoma was about 5%. It was not therefore surprising that the average age in the study was 29.1 years, with a peak in the 10-19year age group. No peripheral ameloblastoma was diagnosed. RESULTS: A total of 47 new cases of ameloblastoma was recorded between 1980 and 1995. This number accounted for 16.03% of all jaw bone lesions in Jamaica and 38.2% of jaw bone lesions of odontogenic origin during this period. These cases of ameloblastoma accounted for 67% of odontogenic tumours with the exception of the odontomas. The mean age was 29.1 (range 13-67) years. The male: female ratio was 1: 1.14). Only 3 cases involved the maxilla. Of significance, is the case of bilateral ameloblastoma with no continuity to the anterior region, and also the case of unilateral involvement of the mandible and the maxilla in the same patient. Reentry cryosurgery (a procedure in which cryosurgery is done after a specified period from the primary surgical procedure) after excisional biopsy of a relatively small cystic ameloblastoma of the maxilla was negative for recurrence. CONCLUSION: The clinicopathological presentation of ameloblastoma in Jamaica is not different from what has been documented by other authors throughout the world; however, we report two unique cases - a case of bilateral ameloblastoma of the mandible and a case of unilateral involvement of the mandible and maxilla in the same patient. We suggest that reentry cryosurgery before recurrence be considered in the management of ameloblastoma for the prevention of recurrence.

OBJETIVO: Analizar todos los casos de ameloblastoma vistos en los dos principales hospitales públicos en Jamaica durante un período de 16 años, y comparar los resultados con la información previamente documentada por otros autores en la literatura. Se describe una nueva modalidad de tratamiento para prevenir la recurrencia del ameloblastoma. SUJETOS Y MÉTODO: Se recuperaron los expedientes de los pacientes diagnosticados histológicamente con ameloblastoma en el Hospital Público de Kingston y el Hospital Regional de Cornwall en Jamaica desde 1980 a 1995, y se obtuvo información documentada acerca de este tumor odontogénico. El análisis reveló que el ameloblastoma unicístico era predominante (95%), en tanto que el ameloblastoma sólido fue de alrededor del 5%. Por lo tanto, no fue sorprendente que la edad promedio en el estudio fuera 29.1 años, con un pico en el grupo etario de 10 a 19 años. No se diagnosticó ningún ameloblastoma periférico. RESULTADOS: Entre 1980 y 1995, se registró un total de 47 nuevos casos de ameloblastoma. Este número representaba el 16.03% de todas las lesiones óseas de mandíbula en Jamaica y 38,2% de las lesiones de hueso de la mandíbula de origen odontogénico durante este período. Estos casos de ameloblastoma representaron el 67% de los tumores odontogénicos con la excepción de los odontomas. La edad promedio fue 29.1 años (rango 13-67). La proporción hombre:mujer fue de 1: 1.14. Sólo tres casos implicaron el maxilar. Resulta particularmente importante el caso del ameloblastoma bilateral sin ninguna continuidad a la región anterior, así como el caso de la participación unilateral de la mandíbula y el maxilar en el mismo paciente. La criocirugía de reentrada (un procedimiento en el cual la criocirugía se realiza después de un período especificado a partir del procedimiento quirúrgico primario) después de la biopsia por escisión de un ameloblastoma quístico del maxilar relativamente pequeño, fue negativa para la recurrencia. CONCLUSIÓN: La manifestación clínicopatológica del ameloblastoma en Jamaica no difiere de lo que ha sido documentado por otros autores en todo el mundo. Sin embargo se reportan dos casos únicos: un caso de ameloblastoma bilateral de la mandíbula y un caso de compromiso unilateral de la mandíbula y el maxilar en el mismo paciente. Sugerimos que se tenga en cuenta la criocirugía de reentrada a la hora de tratar el ameloblastoma para prevenir la recurrencia.

Mineralized components and their interpretation in the histogenesis of peripheral ossifying fibroma.

Background: Peripheral ossifying fibroma (POF) is a lesion of gingival tissue that predominantly affects women and is usually located in maxilla, anterior to molars. The definitive diagnosis is established by histopathological examination, which reveals the presence of cellular connective tissue with focal calcifications. Objective: This study hypothesizes the histogenesis of POF by analyzing the diverse spectrum of mineralized components with a polarizing microscope. Materials and Methods: A retrospective study was undertaken which involved a detailed review of clinical, radiographic and histopathological features of 22 cases of POF, retrieved from departmental archives. These cases were subsequently stained with a histochemical stain (van Gieson) and observed under a polarizing microscope. Results: The study revealed that the most common age of occurrence was in second and third decades with a strong female predilection (73%), Interdental papilla of the maxillary anterior region was the most commonly afflicted site. About 90% cases showed no radiographic features.Histopathological examination showed that 73% cases consisted of a fibrocellular connective tissue stroma surrounding the mineralized masses. 50% mineralized masses comprised of woven bone, 18% showed combination of lamellar bone and cellular cementum, 18% showed only cementum (cellular and acellular), and remaining 13.6% exhibited a mixture of woven and lamellar bone under polarizing microscope. Conclusion: The study supports the theory that POF develops from cells of periodontal ligament (PDL)/periosteum as undifferentiated mesenchymal cells having an inherent proliferative potential to form bone or cementum, whose nature can be confirmed by polarizing microscope.

Adenomatoid odontogenic tumor of the mandible - extrafollicular variety: a rare case report

Adenomatoid odontogenic tumor [AOT] is an uncommon benign odontogenic lesion that affects young patients, with female predominance, mainly in second decade, representing approximately 3% of all odontogenic tumors. It is of interest that in about 74% of cases, this tumor is associated with an unerupted tooth, and in over two third of cases associated with maxillary [65%] or mandibular canine [35%]. Very rarely the lesion occurs without any impacted tooth. In this report a case of adenomatoid odontogenic tumor of right side of mandible in the canine premolar region not associated with an impacted tooth in a 14 years old female patient is described

Systematic review of lymphoma in oral cavity and maxillofacial region

El objetivo del presente trabajo fue determinar las característicasde presentación de pacientes con linfoma en la cavidad oral y la región maxilofacial para comparar estas presentaciones y permitir sintetizar los resultados provenientes de diversos estudios primarios. Fueron incluidos artículos de la base de datos PubMed desde el año 1990 hasta el año 2010. Se incluyeron como palabras claves en el título de los artículos: “Lymphoma”y “Oral”. Se identificaron 215 artículos de los que fueron excluidos 178 estudios quedando para este estudio 37 artículos. Los investigadores evaluaron estos artículos alcanzando un acuerdopara la inclusión de 15 artículos para este estudio. Se describe de los 15 estudios autor, año de publicación del estudio, número de casos, sexo, edad, localización, signos y síntomas, clasificación, diagnóstico, estadiaje y tratamiento. Un total de 714 pacientes se describen siendo en 11 de 13 estudiosel mayor número de casos en hombres que mujeres; la edad estuvo en un rango de 3 a 96 años. La localización de mayorpresentación fue la gingiva y el aumento de volumen fue el principal signo. Solo de 177 casos fueron descritos su estadiaje y solo de 110 casos su tratamiento. Se concluye que los linfomas de la cavidad oral y la región maxilofacial son de mayor presentacióndel tipo no Hodgkin, a nivel extranodal. La gingiva en la cavidad oral y el anillo de Waldeyer en la región maxilofacialson las localizaciones más comunes. Son entidades patológicas raras, pero la descripción de la historia natural deesta patología en su completa manifestación es importante parael conocimiento del desarrollo de esta enfermedad.

Ameloblastomas sólidos: estudo retrospectivo clínico e histopatológico em 54 casos / Solid ameloblastomas: retrospective clinical and histopathologic study of 54 cases

Recentemente, a Organização Mundial da Saúde (OMS) excluiu o padrão desmoplásico do espectro histopatológico do ameloblastomas sólidos e o enquadrou como uma variante distinta, designada ameloblastoma desmoplásico. OBJETIVO: Analisar os achados clínico-patológicos em uma série de casos de ameloblastomas sólidos. FORMA DE ESTUDO: Estudo de coorte transversal. MATERIAL E MÉTODO: Dados sobre idade, sexo, localização e características clínicas foram obtidos em fichas de biópsia. Cortes histológicos foram avaliados quanto aos padrões histológicos existentes e o padrão histológico predominante. Os casos foram classificados com base no estudo de Waldron e El-Mofty (1987) e na classificação da OMS de 2005. RESULTADOS: Foram identificados 54 casos, com distribuição similar entre os sexos e média de idade de 38,3 anos. Cinquenta e três casos (98,1 por cento) afetaram a mandíbula. Quarenta e nove casos (90,8 por cento) foram classificados como ameloblastomas sólidos, 3 (5,6 por cento) como ameloblastomas desmoplásicos e 2 (3,7 por cento) como lesões híbridas. Os padrões histológicos folicular (77,6 por cento), acantomatoso (69,4 por cento) e plexiforme (65,3 por cento) foram os mais frequentes nas lesões sólidas. Áreas focais de ameloblastoma desmoplásico foram identificadas em 11 ameloblastomas sólidos (22,4 por cento). CONCLUSÃO: Embora enquadrado como uma variante distinta, nossos resultados revelam que áreas focais de ameloblastoma desmoplásico podem ser identificadas com relativa frequência em ameloblastomas convencionais.

Recently, the World Health Organization (WHO) excluded the desmoplastic pattern from the histopathological spectrum of solid ameloblastomas and classified it as a distinct variant, named desmoplastic ameloblastoma. AIM: To perform a retrospective analysis of the clinicopathologic aspects in a case series of solid ameloblastomas. STUDY DESIGN: Cross-sectional cohort study. MATERIALS AND METHODS: Data regarding age, gender, location and clinical characteristics were retrieved from patient records. Histological sections were evaluated regarding existing histological patterns and the predominant histological pattern. Cases were classified according to the study of Waldron and El-Mofty (1987) and the WHO classification of 2005. RESULTS: A total of 54 cases were identified, with similar gender distribution and a mean age of 38.3 years. Fifty three cases (98.1 percent) affected the mandible. Forty nine cases (90.8 percent) were classified as solid ameloblastomas, 3 (5.6 percent) as desmoplastic ameloblastomas, and 2 (3.7 percent) as hybrid lesions. The most frequent histological patterns in solid ameloblastomas were follicular (77.6 percent), acanthomatous (69.4 percent), and plexiform (65.3 percent). Focal areas of desmoplastic ameloblastomas were identified in 11 solid ameloblastomas (22.4 percent). CONCLUSION: Despite its characterization as a distinct variant, our results revealed that focal areas of desmoplastic ameloblastomas can be observed with some frequency in conventional ameloblastomas.

Cistos ósseos simples atípicos / Atypical simple bane cysts

O cisto ósseo simples, lesão incomum dentre as patologias da cavidade oral, apresenta uma etiologia ainda inconclusiva, o que se pode constatar frente à variedade de termos que têm sido utilizados para descrever tal condição. Clinicamente, este se apresenta de forma assintomática, sendo na grande maioria dos casos, diagnosticado a partir de um achado ra- diográfico. Apresenta uma predileção pela mandíbula, em regiões dentadas, caracterizando-se radiograficamente como uma lesão unilocular sem expansão das paredes ósseas circundantes. Este artigo visa relatar dois casos clínicos de cistos ósseos simples diagnosticados como atípicos devido ao acometimento do osso mandibular em regiões edêntulas, sendo que em um dos casos apresentou expansão das paredes ósseas; aspecto que, diverge do comumente encontrado na literatura. Ambos os casos clínicos foram submetidos à curetagem e promoção do sangramento das paredes ósseas, com subseqüente formação de um coágulo sanguíneo estável. Como visto na maioria dos casos relatados na literatura, a curetagem da região acometida pelo cisto ósseo traumático apresenta-se como uma alternativa terapêutica extremamente eficaz, devendo o paciente ser periodicamente monitorado, através de exames radiográficos, ate que haja a completa formação do tecido ósseo, já que a recorrência local, apesar de raramente relatada, pode ocorrer.

The sim pie bone cyst is not an ordinary lesion among the oral cavity pathologies and still does not present a conclusive etiology. This can be very well noticed when we face the many terms that have been used to describe it. Clinically speaking, it is not presented in a symptomatic way, being diagnosed, mostly, from X-ray exams. It happens preferably in the teeth regions of the mandible, and can be identified through radiographic exams as an unilocular lesion with no expansion of the surround bone walls. This article aims to relate two clinical cases in which the simple bone cysts have been diagnosed as atypical because of the damage caused on the mandible bone at edentulous regions, considering that in one of them there has been an expansion of the bone walls. Such aspects are not in accordance with what has been described at the common literature. Both clinical cases have been submitted to a curettage proceeding, what developed the bleeding of the bone walls, with subsequent formation of a stable blood coagulum. According to the literature, in most cases, the curettage proceeding at the affected region with the traumatic bone cyst is a very effective therapeutic alternative, in which the patient should be monitored periodically, through radiographic examination, until the bone texture is completely rebuilt, since although it is rare, the local reincidence may occur.

Gnathic osteosarcomas: A 10-year multi-center demographic study.

Background: Osteosarcomas (OS) of the jaws are uncommon lesions representing 6-8% of skeletal OS. We assessed the characteristics, demographics, prevalence clinical and histopathological findings and distribution of gnathic OS relative to non-gnathic OS in four major treatment centers. Materials and Methods: This study assessed 13 gnathic OS patients of 98 OS patients from four major referral centers during 1996-2007. The age distribution, gender, involved site, clinical findings, signs, symptoms, grade and sub-types were assessed. Hematoxylin-eosin, Picrosirius red, Ponceau trichrome, Masson trichrome and osteoid staining methods were used. Results: Of the 98 OS lesions, 85 (86.8%) involved the skeleton, the youngest patient was 6 and the oldest 60 years old; 13 lesions (13.2%) involved the jaws (seven mandibular and six maxillary) and the youngest and oldest patients were 15 and 50 years-old, respectively. Non-gnathic OS was more prevalent between the ages of 11 and 20 years (avg. 15 years) and was common in the distal femur and proximal tibia, presenting most frequently with pain and swelling. OS of the jaws, however, presented more than 10 years later than non-gnathic OS, being more prevalent between the ages of 20 and 30 years (avg. 27 years). OS of the jaws most frequently involved the mandibular body and the posterior maxillary alveolar ridge, presenting frequently with pain, swelling and loosening of teeth. Two patients with gnathic OS died during the 10-year follow-up period (15.3%). Conclusion: Prevalence of OS of the jaws was about twice as high as that reported in other studies and presented later than non-gnathic cases. Pain and swelling were common signs and symptoms in this disease. The mixed sub-type was the most common sub-type of gnathic OS.