Epithelial and Stromal Syndecan-1 roles on the biological behavior of Dentigerous cyst, Ameloblastoma and Ameloblastic Carcinoma

Background: Dentigerous cyst diagnosis is simple but several case reports have documented neoplastic changes arising from them. Ameloblastoma is a common benign Odontogenic tumor with an aggressive manner and a high rate of recurrence. Ameloblastic Carcinoma is the malignant counterpart of Ameloblastoma but usually difficult to be distinguished from each other’s. Hence, need for Immunohistochemical markers may help achievement of accurate diagnosis. Objectives: Evaluation of Epithelial and Stromal Syn1 expressions and their roles in tumorigenesis and biological behavior of Dentigerous cyst, Ameloblastoma and Ameloblastic Carcinoma. Methods: Tissue samples comprising of 54 archived histopathologically confirmed cases of (10 Dentigerous cysts, 29 Ameloblastomas and 15 Ameloblastic Carcinoma). The sections were subjected to Immunohistochemical staining according to a standard protocol using antibody to Syn1. Results: Stromal Syn1 expression was higher in Desmolpastic Ameloblastoma than other Conventional Ameloblastoma subtypes. Unicystic Ameloblastoma showed higher Stromal Syn1 than Dentigerous cyst. Ameloblastic Carcinoma showed the highest immune-reactivity to Stromal Syn1 than Conventional Ameloblastoma. While, Epithelial Syn1 immune-reactivity was weak. Conclusions: Desmoplastic Ameloblastoma behaves in a more aggressive manner than other subtypes.Stromal Syn1 are highly expressed in aggressive and malignant odontogenic tumors and could be used together as prognostic predictor tool for odontogenic tumors

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)

Ameloblastic Carcinoma: A Case Report And Update On Classification System Of Odontogenic Malignancies

Ameloblastic carcinoma is a rare malignant lesion with characteristic behaviour that dictates a more aggressive surgical approach than that of a simple ameloblastoma. However, due to less number of cases reported till now in the literature and scarcity of documentations, reliable evidence of its biologic activity is currently unavailable. Because the lesion is usually found unexpectedly after an incisional biopsy of diagnosed case of Ameloblastoma or after the removal of a cyst, a guide to differential diagnosis is not usually useful. Most ameloblastic carcinomas are presumed to have arisen de novo with a few cases of malignant transformation of ameloblastomas. Although rare, these lesions have been known to metastasize, mostly to the regional lymph nodes or lungs. Here we reported a case of 62year old male and his clinical, radiographical and histological features has been discussed with an insight on the classification systems of odontogenic malignancies.

Sinonasal ameloblastic carcinoma in a 48-year-old Filipino male: A rare fortuity

OBJECTIVE@#To report a case of sinonasal ameloblastic carcinoma in a 48-year-old Filipino male & discuss its clinical manifestations, diagnosis, histopathology and possible treatment options.@*METHODS@#Design: Case Report, Setting: Tertiary Government Hospital, Patient: One@*RESULTS@#A 48-year-old Filipino male consulted at our OPD due to progressive right- sided nasal obstruction, rhinorrhea, hyposmia & intermittent controlled episodes of epistaxis for one year. On anterior & posterior rhinoscopy, a fleshy, soft tissue mass is seen in the nasal floor. On further examination with rigid nasal endoscopy, the same pink, fleshy, soft tissue mass was seen occupying entirely the right nasal floor with nasopharynx hardly to be visualized, and friable upon insinuation. A preoperative punch biopsy of the mass was done at our OPD revealing histologic features consistent with ameloblastoma. Contrast-enchanced computed tomography (CT) scan of the paranasal sinuses revealed a prominent soft tissue density mass lesion filling right sinonasal cavity. It is further noted the heterogenous characteristic of an isodense mass in the right nasal cavity enhancing from the nasopharyngeal area, with complete obstruction of the osteomeatal unit (OMU) and some lytic changes on the medial maxillary sinus wall however no bony changes noted on bilateral orbital floor and skull base. The mass was completely excised by medial maxillectomy via midfacial degloving. Final histopathologic studies showed islands of lace-like areas and nests of atypical odontogenic cells with central stellate reticulum, palisading columnar cells exhibiting reverse polarity and moderate mitotic activity with atypical mitotic figures present on two specimens labelled as "posterior nasal septal mass" & "nasopharyngeal mass".@*CONCLUSION@#Ameloblastic carcinoma is an uncommon entity of malignant odontogenic tumors that may originate de nova or from a benign ameloblastoma which exhibits malignant histologic features in the primary lesion and/or distant metastasis. On the other hand, malignant ameloblastoma exhibit benign histologic fea tures both for the primary and distant metastasis. This report depicted an uncommon case of ameloblastic carcinoma found extragnathically, that is in the sinonasal region. This is the second case of ameloblastic carcinoma of the sinonasal region documented in the Philippines. This case report might be a step on the ladder to generate more information regarding the biologic behavior of this entity and might ignite the enthusiasm in performing more evidence-based studies needed for its treatment as well as for its surveillance. Wide surgical resection, which is the treatment of choice, can pose challenge to otolaryngologist in the case of sinonasal ameloblastic carcinoma due to the surrounding vital structures present. Role of chemotherapy and radiotherapy is as yet conclusive due to very limited evidence- based studies available.

Ameloblastic Carcinoma of Mandible: A Case Report.

Ameloblastic carcinoma is an uncommon malignant odontogenic tumor of jaws and is a highly aggressive tumor with rapid growth and also a high potential for distant metastasis. Th e most common site of occurrence is posterior mandible. Clinically, it is aggressive and has potential for local destruction. Th e majority of the cases arise de novo, while few cases arise from a pre-existing ameloblastoma. Th e radiological feature of the ameloblastic carcinoma is usually similar to that of ameloblastoma except for the presence of focal radiopacity, which apparently refl ects dystrophic calcifi cation. Herein, we report a case of ameloblastic carcinoma in the left mandible of a 60-year-old female patient.

Spindle-cell ameloblastic carcinoma of the maxilla with adenoid cystic carcinoma-like areas: A new variant.

Ameloblastic carcinoma is a rare aggressive malignancy of odontogenic epithelial origin. The disease has been reported to afflict people over a wide range of age group without any sex predilection. It is characterized by rapid growth and destruction of overlying bones along with involvement of adjacent soft tissues. The overall prognosis of this cancer is poor despite radical surgery and radiotherapy. Owing to the rarity of this lesion, complete data on the biological behavior and response to different treatment modalities are still lacking. The authors hereby report a case of this uncommon tumor arising from left maxilla in a 60-year-old Indian male. Histopathological examination of the tumor showed foci of spindling intermixed with large areas of tumor cells arranged in cribriform pattern and having luminal eosinophilic material which was reminiscent of adenoid cystic carcinoma. A final diagnosis was achieved after immunohistochemical study. These unusual findings prompted this case report.

Ameloblastic carcinoma of the mandible.

Ameloblastic carcinoma is a highly malignant tumour and requires aggressive treatment. This case report describes an aggressive ameloblastic carcinoma that infiltrated the mandible. Mandibulectomy with right functional radical neck dissection and left supra omohyoid dissection was followed by primary reconstruction with a single free vascularised fibula flap. The post-operative course was uneventful. The 2 year regular follow up revealed no signs of recurrent tumour or metastasis. Future reporting of this rare condition is encouraged in lieu of limited information in its clinical course and prognosis.

A Case of Ameloblastic Carcinoma in the Maxillary Sinus / 대한이비인후과학회지

Ameloblastoma is the most common benign odontogenic tumor of jaw, whereas ameloblastic carcinoma is extremely rare, aggressive odontogenic neoplasm of mandible and maxilla. Ameloblastic carcinoma is odontogenic malignancy that combines the histological features of ameloblastoma with cytological atypia regardless of metastasis. The clinical course is described as typically aggressive, with extensive local destruction and distant metastatic spread. Ameloblastic carcinoma has been found mostly in the mandible; however, its maxilla lesion has been reported less than 40 cases in the literature. We experienced a case of ameloblastic carcinoma in the maxillary sinus in a 43-year-old male presenting with right facial swelling. No evidence of recurrence was noted at a 5-year follow-up.

Comparative evaluation of expression of proliferating cell nuclear antigen in variants of ameloblastoma and ameloblastic carcinoma.

Aim: To evaluate the use of proliferating cell nuclear antigen index in the different histopathological variants of ameloblastoma, such as the follicular, plexiform, and unicystic types, and in ameloblastic carcinoma by immunohistochemical staining. The proliferating cell nuclear antigen index values of the variants of ameloblastomas and ameloblastic carcinomas are compared in order to determine the biological behavior of these tumors. Materials and Methods: For the present study, archival tissues that had been diagnosed as ameloblastoma and ameloblastic carcinoma were collected from the department of oral pathology. Specimens were embedded in paraffin wax and were sectioned at a thickness of 5 μm and stained with hematoxylin-eosin for reconfirming the histologic pattern. It was also stained immunohistochemically for anti-proliferating cell nuclear antigen antibody. Results: Positive proliferating cell nuclear antigen expression is seen as a light brown, granular stain. The proliferating cell nuclear antigen values of ameloblastic carcinoma were almost five times the value of ameloblastoma. Analysis of variance test, Fischer's exact test/variance ratio test, and Student's t-test were performed and the probability values were determined. Summary and Conclusion: This study showed that ameloblastic carcinoma had the maximum proliferative capacity. Among the variants of ameloblastoma, the plexiform variety had the maximum proliferative capacity, followed by the follicular and unicystic varieties. Altogether, these data indicate that proliferating cell nuclear antigen is related to the biological behavior and proliferation of tumor cells in the variants of ameloblastoma and ameloblastic carcinoma.

Carcinoma ameloblástico / Ameloblastic Carcinoma

Se presenta el caso clínico de una paciente con antecedentes de gastritis, que acudió al consultorio médico de su área de salud por presentar aumento de volumen en paladar, con crecimiento progresivo, acompañado de molestias para masticar y deglutir, la cual fue remitida al Servicio de Cirugía Maxilofacial del Hospital Clinicoquirúrgico Docente "Dr. Joaquín Castillo Duany" de Santiago de Cuba. Los resultados de los exámenes complementarios, incluida la biopsia incisional en paladar, confirmaron que se trataba de un carcinoma ameloblástico. Se indicó seguimiento clínico en el Hospital Oncológico Docente "Conrado Benítez" de la mencionada ciudad.

The case report of a patient with history of gastritis that visited the doctor's office of her health area due to an increase of volume in palate, with progressive growth, accompanied by problems to chew and to swallow is presented. She was referred to the Maxillofacial Surgery Service of "Dr. Joaquín Castillo Duany" Teaching Clinical Surgical Hospital in Santiago de Cuba. The results of the additional tests, including the incisional biopsy in palate, confirmed that it was an ameloblastic carcinoma. Clinical follow-up in "Conrado Benítez" Teaching Oncological Hospital in Santiago de Cuba was suggested.

Bone scintigragphy in pediatric patient with maxillary ameloblastic carcinoma

Presented here is a rare occurrence of ameloblastic carcinoma located in the maxilla of a pediatric patient. The patient's chief complaint is a large mass protruding from the right maxillary area diagnosed as ameloblastic carcinoma before consulting the nuclear medicine section for whole-body bone scintigraphy. Whole-body bone scintigraphy was performed with cranial SPECT imaging. The maxillary tumor appeared to have decreased radiotracer uptake with intense activity in the anterior and anteromedial aspects of the tumor. There was also a focus of uptake in the lateral border of the tumor, radiating to the angle of the right mandible. Mandibular X-ray series was also acquired, with findings that correlate with those of the bone scintigraphy.

Differentiation and proliferation of ameloblastic carcinoma : immunohistochemical study using Ki-67, CK-LM and CK-HM antibody

A Case Report Of Ameloblastic Carcinoma On The Mandible

Carcinomas derived from ameloblastomas have been designated by a variety of terms, including malignant ameloblastoma, ameloblastic carcinoma, metastatic ameloblastoma, and primary intra-alveolar epidermoid carcinoma. The term of ameloblastic carcinoma is differentiated from the term of malignant amelblastoma and is defined as an ameloblastoma in which there is histologic evidence of malignancy in the primary tumor or the recurrent tumor(or metastasis), regardless of whether it has metastasized. The well-documented and adequately followed cases are currently lacking and this report described an instance of ameloblastic carcinoma with good result after treatment and review of literature.