Giant Mandibular Ameloblastoma with Rare Hypercalcemia: A Case Report and Literature Review.

Ameloblastoma is the most common benign odontogenic tumor with local invasion and high recurrence, which generally occurs in the jaw bones. Hypercalcemia is a common paraneoplastic syndrome that is commonly observed in patients with malignancies but rarely encountered in patients with benign tumors. Thus far, not many cases of ameloblastoma with hypercalcemia have been reported, and the pathogenic mechanism has not been studied in depth. This paper presents a case report of a 26-year-old male diagnosed with giant ameloblastoma of the mandible, accompanied by rare hypercalcemia. Additionally, a review of the relevant literature is conducted. This patient initially underwent marsupialization, yet this treatment was not effective, which indicated that the selection of the appropriate operation is of prime importance for improving the prognosis of patients with ameloblastoma. The tumor not only failed to shrink but gradually increased in size, accompanied by multiple complications including hypercalcemia, renal dysfunction, anemia, and cachexia. Due to the contradiction between the necessity of tumor resection and the patient's poor systemic condition, we implemented a multi-disciplinary team (MDT) meeting to better evaluate this patient's condition and design an individualized treatment strategy. The patient subsequently received a variety of interventions to improve the general conditions until he could tolerate surgery, and finally underwent the successful resection of giant ameloblastoma and reconstruction with vascularized fibular flap. No tumor recurrence or distance metastasis was observed during 5 years of follow-up. Additionally, the absence of hypercalcemia recurrence was also noted.

Transformation of ameloblastoma to ameloblastic carcinoma in a 10-year-old child.

Ameloblastic carcinoma (AC) is a rare odontogenic malignant epithelial neoplasm of maxillofacial skeleton with a distinct predisposition of the mandible. It can occur in a wide range of age groups, with a sex predilection in males. It can arise either as a de novo lesion or from preexisting ameloblastoma. AC has a high propensity for local recurrence as well as distant metastasis (chiefly lungs), thus requiring an aggressive surgical approach and a strict surveillance. Owing to the rarity of publications describing AC, little is known about this entity in pediatric patients. We report a case of transformation of ameloblastoma into AC in a 10-year-old child.

A rare case report of extensive mandibular osteoma corrected by unilateral mandibulectomy: Cytological, radiological, and pathological investigation.

Background: Osteoma is a benign bone tumor that rarely affects animals. The most common bones involved with this tumor included the mandible, maxillofacial bones, and nasal sinuses. Definitive diagnosis is based on pathology findings which allow for differentiation with other bone lesions. Case Description: The patient, a 5-year-old intact male Mongrel dog presented with a huge mandibular mass that involved both the right and left mandible, and led to dental occlusion. The radiography was performed and depicted the intense mass with a well-demarcated edge, a short transitional zone between normal and abnormal bone, and a smooth rounded radiopaque appearance. The investigation according to the fine needle aspiration showed the presence of oval to spindle shape cells with poorly malignancy criteria, fatty cells, reactive osteoblasts, and osteoclasts based on a population of spindle-shaped cells, and low numbers of degenerated neutrophils, bacteria, and few macrophages. Then, the radiographic assessments and cytology findings demonstrated the osteoma and were referred for surgical intervention. A unilateral mandibulectomy was performed, and the lesion was sent to the histopathology laboratory. The histopathology evaluation showed osteocyte proliferation without malignancy features. The osteoblast cells also showed no atypical proliferation that endorses the osteoma tumor. Conclusion: Although mandibular and maxillofacial bone resection in small animals have different tolerations, this patient became a candidate for surgery for future better nutrition and prevention of facial deformity and dental malocclusion. Follow-up after osteoma is one of the most necessary post-operation treatments to check the regeneration of the mass. There are considerable data in this report that should regard this tumor as a possible differential diagnosis for mandibular tumors.

A multi-disciplinary team approach to pediatric malignant mandibular tumors.

OBJECTIVE: Mandibular tumors in the pediatric population are rare. These malignancies are variable in their histology, and combined with their rarity, has made it difficult to describe their clinical course, and treatment guidelines. The aim of this paper is to describe the experience of Boston Children's Hospital, a pediatric tertiary referral center, with treating malignant mandibular malignancies, as well as provide multi-disciplinary team approach in managing this clinical entity. METHODS: A retrospective search was performed for mandibular malignancies in pediatric patients between 1995 and 2020 via the pathological database at Boston Children's Hospital. Only patients with malignant solid mandibular neoplasms were included, leaving 15 patients for final analysis. RESULTS: The median age at presentation was 10.1 ± 10.3 years. Nine of 15 patients (60%) presented with jaw mass which was the most common clinical presentation. The most commonly identified histological diagnosis was rhabdomayosarcoma and osteosarcoma (n = 4, 26% each). A mandibulectomy was performed in 12 (80%) cases. Reconstruction of the mandible was performed using a fibular free flap in 6 (40%) cases, and a plate in 3 (20%) cases. Mean follow-up was 4.6 ± 4.9 years. CONCLUSION: Malignant tumors most commonly present with a jaw mass, however asymptomatic and incidental presentations follow closely and pathologies can vary greatly. Surgical resection and reconstruction is often indicated, multidisciplinary tumor board review is required to determine when children are best treated with neo-/adjuvant treatment with chemo- and radiotherapy.

Ameloblastic Fibro-Odontoma.

Introduction: Ameloblastic fibro-odontoma (AFO) is a benign odontogentic tumor without an aggressive behavior, unlike the similar ameloblastic fibroma. Case Presentation: A 9-year-old boy, with tooth eruption failure, underwent enucleation and curettage of a well-defined variable radiolucent and radio-opaque right mandible lesion. There was odontogenic epithelium with peripheral palisading in a loose myxoid stroma as well as a disorganized component of dentin, enamel, and cementum, features of an AFO. Conclusion: AFO is an odontogenic mixed tumor of epithelium and mesenchyme.

A Recurrent Case of Ameloblastic Fibroma in 37-year Old Male.

Ameloblastic fibroma (AF) is a benign mixed epithelial and mesenchymal odontogenic tumor. This was previously grouped in odontogenic tumor showing odontogenic epithelium with odontogenic ectomesenchyme, with or without hard tissue formation. This report describes a case of ameloblastic fibroma in a 37-yearold male who came with the complain of swelling in the left side of lower jaw since one year. Enucleation of the mass followed by reconstruction was done six years back. However, after two years of initial treatment; radiographic findings suggested recurrence. Histopathological examination confirmed the diagnosis of ameloblastic fibroma. Patient had no clinical and radiographic evidence of recurrence in three and six months' follow-up. Because of the higher proliferative capacity and malignant degree of the mesenchymal component in the recurrent neoplasm, sarcomatous transformation may occur. Hence, a long term clinical and radiographical follow-up is essential due to its transformation into ameloblastic fibrosarcoma.

Life-threatening mandibular angle gigantic osteoma presenting with severe dyspnoea.

Osteoma is a benign osteogenic tumour. Solitary osteoma of the jaws usually involves the mandible and commonly remains asymptomatic. Purpose of this article is to report a case of life-threatening gigantic mandibular osteoma in an edentulous woman in her 70s developed in the lingual side of the mandibular angle presenting at emergency department with dyspnoea and discuss the correct management of the patient and the surgical approach for space occupying mass in the pharapharyngeal space.

Hypoproteinemia Associated with a Gigantic Odontogenic Tumor: A Report of 2 Cases.

BACKGROUND Hypoproteinemia is caused by a decrease in protein level in the blood. This report describes 2 cases of hypoproteinemia associated with a gigantic odontogenic tumor. CASE REPORT Case 1, a 65-year-old man, visited our hospital with the chief concern of swelling in the right mandible, approximately 100 mm in diameter, and ameloblastoma was diagnosed. Abscess drainage was observed in the fistula of the tumors. Total protein and albumin levels were low before surgery. Hemimandibulectomy was performed under general anesthesia. The final pathological diagnosis based on the specimen was ameloblastic carcinoma. After surgery, the total protein and albumin levels improved and remained stable 6 months after the operation. At 21 months after surgery, there were no signs of recurrence. Case 2, a 60-year-old woman, visited our hospital with a chief concern of swelling in the left mandible, approximately 100 mm in diameter, and ameloblastoma was diagnosed. Abscess drainage was observed in the fistula of the tumors. The patient had a history of hypoproteinemia; preoperative levels of total protein and albumin were low, and edema of the body was observed before surgery. Hemimandibulectomy was performed under general anesthesia. The final pathological diagnosis based on the specimen was ameloblastoma. After surgery, the total protein and albumin levels improved, and remained stable 6 weeks after surgery. There were no signs of recurrence 9 months after surgery. CONCLUSIONS These 2 cases indicate the possibility that hypoproteinemia can be caused by plasma leakage from fistulas associated with gigantic odontogenic tumors.

Fibroma ossificante juvenil trabecular: relato de caso / Juvenile trabecular ossifying fibroma: case report / Fibroma osificante trabecular juvenil: reporte de un caso

O fibroma ossificante juvenil trabecular (FOJTr) é uma lesão fibro-óssea benigna rara de comportamento agressivo, alto potencial de recorrência, e acometimento no esqueleto craniofacial de crianças e adolescentes. Uma paciente do gênero feminino, 8 anos de idade, compareceu ao ambulatório de Patologia Oral e Maxilofacial da Universidade de Gurupi ­ UNIRG para avaliação clínica de um aumento de volume na região de corpo da mandíbula do lado esquerdo. Não havia sintomatologia dolorosa e sequer desconforto. Nos exames de imagem (radiografia panorâmica e tomografia computadorizada) foram observados uma extensa área radiolúcida que se estendia desde o primeiro molar permanente com rizogênese incompleta até o incisivo central do lado oposto. Após a realização da biópsia incisional e laudos histopatológicos realizou-se a remoção completa da lesão incluindo os remanescentes decíduos sobrejacentes ao fibroma. Nas imagens de controle pós-operatório aos 90 dias (radiografia panorâmica e tomografia computadorizada), notou-se sinais de neoformação óssea com espessamento basilar e os germes dos dentes permanentes em franco desenvolvimento. Diante disso, ressalta-se a importância do conhecimento dos aspectos clínicos, radiográficos e histopatológicos para a realização de um correto diagnóstico e tratamento adequado afim de reduzir as altas taxas de recidivas... (AU)

Trabecular juvenile ossifying fibroma (TrJOF) is a rare benign fibro-osseous lesion, with aggressive behavior, high recurrence potential, which affects the craniofacial skeleton of children and adolescents. This paper aims to describe a clinical case in a female patient, 8 years old, who attended the Oral and Maxillofacial Pathology outpatient clinic ­ Faculty of Dentistry ­ University of Gurupi - UNIRG, city of Gurupi - TOCANTINS - BRAZIL for clinical evaluation of an increased in volume in the region of the mandible body, on the left side. There was no painful symptomatology or even discomfort. Imaging examinations (panoramic radiography and computed tomography (CT) showed an extensive radiolucent area that extended from the first permanent molar with incomplete root formation to the central incisor on the opposite side. After performing an incisional biopsy and histopathological examination, the lesion was completely removed included the remainder deciduous teeth overlying the tumor. In the postoperative control images at 90 days (panoramic radiography and CT), signs of bone neoformation with basilar thickening and the germs of the permanent teeth in full development were noted. In view, this importance of knowledge of clinical, radiographic and histopathological aspects is emphasized for the realization of a correct diagnosis and adequate treatment in order to reduce the high rates of relapses... (AU)

El fibroma osificante trabecular juvenil (TRFOJ) es una lesión fibroósea benigna rara con comportamiento agresivo, alto potencial de recurrencia y afectación del esqueleto craneofacial de niños y adolescentes. Paciente femenina de 8 años de edad que acude al ambulatorio de Patología Oral y Maxilofacial de la Universidad de Gurupi - UNIRG para evaluación clínica de aumento de volumen en la región del cuerpo mandibular del lado izquierdo. No presentaba sintomatología dolorosa ni molestias. Los exámenes de imagen (radiografía panorámica y tomografía computarizada) mostraron una extensa área radiolúcida que se extendía desde el primer molar permanente con formación radicular incompleta hasta el incisivo central del lado opuesto. Tras realizar la biopsia incisional y los informes histopatológicos, se procedió a la extirpación total de la lesión, incluidos los remanentes caducos que recubrían el fibroma. En las imágenes de control postoperatorio a los 90 días (radiografía panorámica y tomografía computarizada), se observaron signos de neoformación ósea con engrosamiento basilar y los gérmenes de los dientes permanentes en pleno desarrollo. Por tanto, es importante conocer los aspectos clínicos, radiográficos e histopatológicos para la realización de un diagnóstico correcto y un tratamiento adecuado con el fin de reducir las altas tasas de recaídas... (AU)

Recurrence of ameloblastoma in bone grafts of a fibula free flap: A case report and literature review.

Ameloblastoma is a benign odontogenic tumor with high recurrence rate. It can cause severe abnormalities of the face and jaw, leading to severe disfiguration and impaired jaw and airway functions. Wide local excision and reconstruction are required for ameloblastoma. Recurrence in the free bone graft is rare, especially in the fibula free flap. A 67-year-old man had a history of ameloblastoma of the left mandible resected 16 years ago with reconstruction using the fibula free flap. Three years ago, radiologic examination suggested a possible tumor recurrence in the fibula free flap. Because of the left facial swelling with hard, painless, and elastic tumor extending in the mandibular, he was admitted to the hospital and underwent segmental mandibulectomy. Ameloblastoma in the fibula was confirmed by pathological examination meanwhile not invasion in soft tissue nor bone margin. At the 24-month follow-up, no recurrence was detected. As the strong tendency for recurrence, especially in cases of large tumor size and soft tissue involvement, the radical resection and long-term follow-up are recommended.

Ameloblastic carcinoma in horses: case report and literature review.

Ameloblastic carcinoma is a malignant odontogenic neoplasm that has been reported only rarely in veterinary species. A 16-y-old Arabian crossbred mare was presented for evaluation of a hard mass on the body of the mandible, with evidence of osteolysis on radiographs. Incisional biopsies revealed an invasive neoplasm comprised of spindloid epithelial cells with a high mitotic count and partial dual cytokeratin-vimentin immunoreactivity. The horse was euthanized because of rapid tumor progression 3 mo after presentation. Postmortem evaluation revealed partial obliteration of the mandible by a large, firm-to-hard, tan, locally destructive and invasive mass with no gross or histologic evidence of metastasis. Postmortem histology revealed a poorly differentiated epithelial neoplasm with variably prominent features suggestive of odontogenic histogenesis: a plexiform ribbon architecture, infrequent basilar palisading with antibasilar nuclei, rare basilar cytoplasmic clearing, subepithelial matrix hyalinization, and partial dual cytokeratin-vimentin immunoreactivity. Features of malignancy included regions of necrosis, pronounced cellular atypia, a high mitotic count, extensive tissue invasion and local tissue destruction, and extension of neoplastic cells beyond the margins of the mandibular bone. Collectively, these features are most consistent with mandibular ameloblastic carcinoma. Including our case described here, ameloblastic carcinoma has been reported in only 5 horses. The microscopic features reported most consistently are dual cytokeratin-vimentin immunoreactivity, a high mitotic count, and basilar palisading. Ameloblastic carcinoma should be considered as a differential diagnosis for rapidly growing, locally invasive masses arising from the dentate jaw of horses.

Peripheral ameloblastic fibro-odontoma-Report of 2 cases of a very rare entity.

Ameloblastic fibro-odontoma (AFO) is a rare benign mixed odontogenic tumor that affects children and young adults. AFO occurs mainly intraosseous. Extraosseous AFO is extremely rare. We report 2 cases of rare peripheral ameloblastic fibro-odontoma in 2- and 12-year-old female patients. Microscopic examination revealed a benign proliferation of odontogenic epithelium associated with a dentinoid material distributed within a cell-rich mesenchymal stroma resembling dental papilla. Simple surgical excision of the lesion is usually curative. There was no recurrence after a short period of follow-up. Clinicians should be cognizant of this rare entity, which can be considered in a differential diagnosis of gingival growths that are noted in early childhood.

Rare clear cell odontogenic carcinoma associated with impacted tooth in a young patient: case report and literature review.

Clear cell odontogenic carcinoma (CCOC) is a rare malignant odontogenic tumor. It is characterized by showing, on histopathological examination, clusters of vacuolated and clear tumor cells with epithelial differentiation surrounded by fibrocollagenous stroma and fibroblasts. The present study presents a rare clinical case of mandibular CCOC associated with an impacted tooth in a 26-year-old woman surgically treated with mandibulectomy and reconstruction with iliac crest bone graft. The patient has been followed up for 22 months without signs of recurrence. A search for case report/case series was carried out in the PUBMED database, as well as in the references of relevant previously published literature reviews. Ninety-six publications were identified, totaling 136 distinct cases reported. Female sex was the most affected (63.1%) with 63.3% of cases occurring in patients in the fifth, sixth, or seventh decades of life. The mandible was more affected than the maxilla (74.2%). Association of CCOC with impacted teeth was found in 2.4% of cases, thus rendering it a rare occurrence. The present case report corroborates the results of the survey regarding sex and anatomical location of the tumor; however, it contradicts the findings regarding age predilection. The case described is the fourth known occurrence of tooth impaction associated with the tumor and the first in a female. In conclusion, CCOC should be considered, as well as other malignancies, as a possible diagnosis of maxillary or mandibular intraosseous lesions even in unusual circumstances such as in association with impacted teeth and in young patients.

Melanotic neuroectodermal tumor of infancy in the mandible: A case report.

RATIONALE: Melanocytic neuroectodermal tumor of infancy (MNTI) is a rare benign pigmented neoplasm that arises from the neural crest and has an aggressive growth pattern. It is predominantly seen in infants under 1 year of age, and the most common site of involvement is the maxilla. The currently accepted treatment is removal by surgical resection. Herein, we report a case of MNTI that involved the anterior alveolar ridge of the mandible in a 6-month-old infant. PATIENT CONCERNS: A case of a 6-month-old male child with a huge mass in the anterior alveolar ridge of the mandible. DIAGNOSIS: The tumor was diagnosed using histopathological and immunohistochemical techniques on the biopsy specimen obtained following incisional biopsy. Based on the findings, a final diagnosis of MNTI was established. INTERVENTIONS: Radical resection of the tumor was performed, after determining the extent of resection by referring to the mandibular 3D model created using the pre-operative CT data. OUTCOMES: The postoperative course was uneventful, and no recurrence has been observed to date for more than 4 years after surgery. LESSONS: This case emphasizes that early diagnosis and radical surgery are critical to the effective treatment, as MNTI exhibits rapid and destructive growth. It also requires careful and close follow-up because of high recurrence rates.

Intrabony mandibular metastases caused by papillary thyroid carcinoma masquerading as a parotid swelling.

Papillary carcinoma is the most common type of thyroid cancer, accounting for 80-90% of cases. Distant metastasis of papillary thyroid cancer is uncommon, but when it does occur, it is most commonly to the lungs, liver and bone. Bone metastases to the mandible are rarely reported. We present a 68-year-old man who was referred due to a right parotid mass. Appropriate imaging and biopsy revealed a thyroid malignancy with bone metastases. The patient subsequently underwent thyroidectomy, with histology revealing multifocal papillary carcinoma. Radioactive iodine treatment was then commenced to control the metastatic disease.

[Adenomatoid odontogenic tumor: about two cases]. / La tumeur adénomatoïde odontogène: à propos de deux observations.

Adenomatoid odontogenic tumor is a benign epithelial tumor which mainly affects young women. It usually occurs in the anterior portion of the upper jaw. Diagnosis can be clinically suspected in patients with cyst formation associated with retained tooth but histopathological confirmation is required. This study involved two patients aged 13 and 37 years, with no previous history, presenting after the occurrence of a swelling on the jaw and mandible. Anatomopathological examination of these lesions showed adenomatoid odontogenic tumor. This study highlights the anatomoclinical features, outcome and treatment of this type of tumor.