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.

Giant Aggressive Odontogenic Fibroma in a 15-month-old Female: Case Report and Review of Literature.

INTRODUCTION: Odontogenic fibroma (OF) is a rare benign tumor of mesenchymal odontogenic tissue with variable global incidence. Two variants comprising extraosseous /peripheral (POF) and intraosseous /central (COF) have been identified based on the anatomical origin of the tumor. OF isslow growing, and often asymptomatic with characteristic radiographic appearance of a unilocular or multilocular cyst frequently associated with unerupted or displaced teeth while histologically, varying amount of inactive odontogenic epithelium embedded in a mature relatively dense collagenous stroma is diagnostic. The absence of odontogenic epithelium does not exclude the diagnosis of odontogenic fibroma. CASE REPORT: A 15 months old female presented with 3 months history of left lower jaw swelling which showed aggressive growth following incisional biopsy. Parents said lesion was noticed for the first time as pinpoint on the submandibular area and was too small to warrant any worries. Initial radiograph revealed absence of bony involvement but the second showed evidence of bony involvement. An impression of soft tissue sarcoma was made and incisional biopsy was sent for pathological diagnosis which revealed peripheral odontogenic fibroma. The rapidity of growth following incisional biopsy bellied a benign lesion. CONCLUSION: An aggressive odontogenic fibroma with multifocal points in a 15months old girl.

INTRODUCTION: Le fibrome odontogène (FO) est une tumeur bénigne rare du tissu odontogène mésenchymateu dont l'incidence globale est variable. Deux variantes comprenant des fibromes extra-osseux /périphérique (POF) et intra-osseuse / centrale (COF) ont été identifiés en fonction de l'origine anatomique de la tumeur. La POF est à croissance lente, et souvent asymptomatique avec l'apparence radiographique caractéristique d'un kyste uniloculaire ou multiloculaire fréquemment associé à des dents non érigées ou déplacées alors que histologiquement, une quantité variable d'épithélium inactif noyé dans un stroma collagène mature relativement dense est le diagnostic. L'absence d'épithélium odontogène n'exclut pas le diagnostic de fibrome odontogène. RAPPORT DE CAS: Une femme de 15 mois a présenté un gonflement de la mâchoire inférieure gauche depuis 3 mois de gonflement de la mâchoire inférieure gauche qui a montré une croissance agressive après une biopsie incisionnelle. Les parents ont déclaré que la lésion avait été remarquée pour la première fois sur forme de point d'épingle dans la zone submandibulaire et était trop petite pour justifier une quelconque inquiétude. La radiographie initiale a révélé l'absence d'atteinte osseuse mais la seconde a montré des signes d'atteinte osseuse. Une impression de sarcome des tissus mous et une biopsie incisionnelle a été envoyée pour un diagnostic pathologique qui a révélé un fibrome odontogène périphérique. La rapidité de la croissance après la biopsie incisionnelle a fait croire à une lésion bénigne. CONCLUSION: Un fibrome odontogène agressif avec des points multifocaux chez une jeune fille de 15 mois. Mots clés: Fibrome odontogène, Périphérique, Central, Agressif.

Odontogenic fibroma amyloid-variant: a typical case and brief considerations about mimickers.

The World Health Organization's (WHO) updated classification of head and neck tumors (2017) defined odontogenic fibroma as a rare neoplasm. In this report, we describe an unusual, typical and rare variant of a central odontogenic fibroma with diffuse amyloid-like protein stromal deposition, and discuss the differential diagnosis with other entities. Radiographically, this lesion presented as a well-defined radiolucency of the mandible, partially cystic. Histologically, the lesion showed a unique confluence of odontogenic epithelial rests in a moderately cellular connective tissue. Immunohistochemical staining highlighted a mixture of benign epithelial and Langerhans cells within connective tissue with diffuse amyloid-like stromal deposition. The importance of recognizing this variant of odontogenic fibroma is due to its benign prognosis and clinical course.

Chondromyxoid fibroma-like osteosarcoma: a case series and literature review.

BACKGROUND: Chondromyxoid fibroma-like osteosarcoma (CMF-OS) is an exceedingly rare subtype of low-grade central osteosarcoma (LGCO), accounting for up to 10% of cases and making it difficult to diagnose. CMF-OS is frequently misdiagnosed on a radiological examination and biopsy, even after the initial operation. Its treatment is a controversial issue due to its low-grade classification and actual high-grade behavior. CASE PRESENTATION: We retrospectively reviewed the medical charts of more than 2000 osteosarcoma patients between 2008 and 2019; 11 patients with CMF-OS were identified, of which six patients were treated by our institution with complete clinical characteristics, including treatment and prognosis, radiological and pathological features were reviewed. Three males and three females with a median age of 46 (range 22-56) years were pathologically proven to have CMF-OS. The radiological presentation of CMF-OS is variable, thus radiological misdiagnoses are common. However, one must not ignore a malignant radiologic appearance. The most distinctive pathological feature conferring the diagnosis of CMF-OS is the presence of osteoid production directly by the tumor cells under a chondromyxoid fibroma (CMF)-like background. Differential diagnoses based on comprehensive data from CMF, LGCO, chondrosarcoma (CHS), conventional osteosarcoma (COS), etc., are needed. All patients were treated with an operation and chemotherapy, and one patient received additional radiotherapy. Nevertheless, recurrence and metastasis are common in CMF-OS patients. Relatively invasive biological behavior of CMF-OS is against the low-grade classification of this disease. CONCLUSIONS: It is important to recognize CMF-OS and distinguish it from CMF, CHS, COS and other LGCOs. CMF-OS has a relatively poor prognosis despite its low-grade classification.

Paediatric oral surgery: endoscopic approach in ameloblastic fibroma management. A preliminary report.

AIM: Ameloblastic fibroma (AF) is a rare benign odontogenic tumor of slow-growing behaviour characterised by proliferation of both epithelial and mesenchymal odontogenic tissues. Treatment of AF is usually conservative by means of enucleation, curettage and long term follow-up. CASE REPORT: This report describes a case of AF in a 12-year-old boy. Examination of the oral cavity revealed absence of tooth 3.7 and mild swelling in the same mandibular area. Orthopantomography and Cone Beam Computed Tomography (CBCT) showed a wide multilocular radiolucent lesion at the left mandibular angle, extending from the first molar to the lower part of the mandibular branch; agenesis of tooth 3.7 and displacement of tooth 3.8. Surgical excision was performed under general anaesthesia. A fiberscope was used to perform a more conservative bone removal and extraction of tooth 3.8. Furthermore, an accurate curettage of the bone site was performed. The histological investigation showed an AF pattern: an epithelial cell component arranged in nests and tubules immersed in a stroma of low differentiated mesenchymal tissue. No sign of recurrence has been observed during the 6-month follow-up period. CONCLUSION: Although AF is a rare tumor, it is more prevalent in children's jaw. A new intra-operative visual technique could provide a conservative treatment by minimising bone deformities and permitting an accurate bone curettage.

Fibromatosis associated with silicone breast implant: ultrasonography and MR imaging findings.

Desmoid type fibromatosis is an uncommon benign disease entity of which its etiology is currently unknown. It constitutes 0.3% of all solid neoplasms, but it is rarely seen in the breast and even more scarcely reported to develop in association with breast implant. We present ultrasonography and magnetic resonance imaging findings of a 29-year-old female patient with fibromatosis after breast implant surgery. Knowledge of imaging findings of breast fibromatosis associated with implant will be helpful for accurate diagnosis and appropriate management.

Arthropathy, osteolysis, keloids, relapsing conjunctival pannus and gingival overgrowth: a variant of polyfibromatosis?

Polyfibromatosis is a rare fibrosing condition characterized by fibromatosis in different body areas and by keloid formation, and which can be associated with arthropathy and osteolysis. Familial occurrence has been described, but the cause remains unknown. Here, we describe a patient with characteristics of polyfibromatosis with arthropathy who had in addition severe conjunctival fibrosis, distinctive face, gingival overgrowth, and pigmented keloids. We discuss the resemblances and differences with polyfibromatosis and descriptions of other, similar patients. We conclude that at present it remains uncertain whether the patient has a variant of polyfibromatosis or a separate entity.

Ameloblastic fibroma diagnosis, treatment and propensity for misidentification.

We present a case of a young male with a radiolucency of the posterior mandible that was diagnosed as an ameloblastic fibroma, a mixed lesion derived from odontogenic epithelium and ectomesenchyme. This case report highlights a potential pitfall of this entity that may lead to inaccurate identification and treatment. Initial frozen section analysis yielded a diagnosis of odontogenic myxoma due to histologic similarities. Our report and discussion help to reacquaint the general practitioner with this entity and emphasize the need for routine screening radiographs and close clinical correlation whenever a microscopic diagnosis is rendered.

Central odontogenic fibroma with amyloid: a diagnostically challenging case.

Odontogenic fibroma is a rare benign mesenchymal odontogenic tumor, with its histological diversity possibly posing diagnostic challenges. A case of the amyloid variant of central odontogenic fibroma, with epithelial cells in perineural and intraneural locations, is reported herein. The 46-year-old female patient had experienced discomfort related to her anterior right hard palate for approximately 25 years. Clinical examination revealed a depression in the anterior hard palate, and radiographic examination showed a well-defined radiolucent lesion with root resorption of the adjacent teeth. Histologically, the well-circumscribed tumor was composed of hypocellular collagenous connective tissue with small islands of odontogenic epithelium. In addition, the juxta-epithelial deposition of amyloid globules without calcification and epithelial cells in perineural and intraneural locations were observed, which posed a diagnostic challenge in differentiating the lesion from the non-calcifying variant of calcifying epithelial odontogenic tumor and sclerosing odontogenic carcinoma. However, on the basis of the clinical and radiographic findings, which were suggestive of a benign and slowly progressive process given the corticated, unilocular radiolucency, the considerable root resorption, and the long history of this finding in an otherwise healthy patient, the final diagnosis was amyloid variant of central odontogenic fibroma. Increased recognition of this variant of odontogenic fibroma and its differentiation from other more aggressive lesions could help the clinician to avoid overdiagnosis and overtreatment.

Rare hybrid tumor of odontogenic fibromyxoma and central giant cell granuloma in maxilla: First reported case.

Fibromyxoma is a locally aggressive rare benign tumor of mesenchymal origin with or without odontogenic epithelium. The etiology of this tumor remains unknown and it is responsible for approximately 3-8% of all cysts and tumors. Another locally destructive benign lesion is central giant cell granuloma (CGCG) which contains osteoclast-like multinucleated giant cells. CGCG accounts for about 7% of all benign jaw tumors, which usually affects younger females. A hybrid lesion with histologic features of both central fibromyxoma and CGCG has not been reported in the literature so far. In the present article, we report the first case of a hybrid tumor comprising odontogenic fibromyxoma with CGCG in a female along with a brief review of its clinical presentation, radiographic features, histological features, and management.

Central odontogenic fibroma in association with brown tumor of hyperparathyroidism in a patient with neurofibromatosis type 1.

We present a patient with bone abnormalities and a myriad of lesions secondary to his redeveloping renal failure and neurofibromatosis type 1 (NF1). A 21-year-old male renal transplant recipient with NF1 presented with painless masses and large, irregular radiolucent lesions in the maxilla and mandible. After histologic examination, the lesion was diagnosed as a central odontogenic fibroma (COdF) in association with a central giant cell lesion, most consistent with brown tumor of hyperparathyroidism. The bone changes were interpreted to be highly suggestive of renal osteodystrophy. Around 30 cases of hybrid central giant cell granuloma-like lesion in association with central odontogenic fibroma have been reported. This, to our knowledge, is the first reported case of brown tumor in association with COdF. Our case provides further evidence of the giant cell component as the initiating entity in these hybrid lesions.

Nonossifying fibroma secondary to aneurysmal bone cyst in the mandibular condyle.

Nonossifying fibromas (NOFs) are benign lesions that unusually occur in the mandible. Nonossifying fibromas are asymptomatic and spontaneous resolution at skeletal maturity. Nonossifying fibromas associated with aneurysmal bone cyst (ABC) are very rare. In this clinical report, NOF secondary to ABC in the mandibular condyle was reported; however, it presents different clinical behavior than the usual NOF. In this case, severe destruction in the mandibular condyle as a characteristic of NOF was seen. In the follow-up period, no recurrence was seen subsequent to treatment of lesion with complete resection. Treatment of NOFs with secondary ABC would require aggressive intervention than the treatment of usual NOF.

Peripheral odontogenic fibroma associated with a dilacerated maxillary central incisor: a case report.

The authors report a case of a 16-year-old male with peripheral odontogenic fibroma (POF) in the anterior maxilla associated with dilaceration of a tooth in its vicinity. A solitary, exophytic and sessile growth was present between the maxillary right central and lateral incisors and extended from the labial mucosa to the palatal gingiva. A periapical radiograph of the maxillary right central incisor revealed a shortened and dilacerated root. The growth was excised and sent for histopathologic examination. A diagnosis of POF (World Health Organization type) was rendered. The clinical and microscopic features are discussed.

Severe presentation of non-ossifying fibroma of the femur in osteoglophonic dysplasia.

Osteoglophonic dwarfism, also known as osteoglophonic dysplasia (OD), is an uncommon skeletal dysplasia with an autosomal dominant mode of inheritance, which equally affects boys and girls. OD is saliently featured by craniosynostosis, dysmorphic facial features, impacted mandibular teeth, rhizomelic limb shortening and non-ossifying fibromas habitually at the metaphyseal regions, which usually disappear after skeletal maturity. The long bones in OD are portrayed by this distinguishable 'hollowed-out' appearance with metaphyseal cystic defects that have a natural history of spontaneous resolution. We report a case of a rare and unusual presentation of OD in a 23-year-old woman whom has been diagnosed with OD during her early childhood. She presented with a progressively enlarging right thigh swelling associated with pain for the past 1 year. Her right femur plain radiograph revealed diffuse lysis of the whole femur with cortical thinning. MRI revealed multiple bilateral femur benign cystic lesion synonymous with a severe spectrum of OD. She was started on a trial of oral bisphosphonates, which led to a significant improvement in pain.

Central odontogenic fibroma: an international multicentric study of 62 cases.

OBJECTIVE: The aim of this study was to report the clinicopathologic features of 62 cases of central odontogenic fibroma (COdF). STUDY DESIGN: Clinical and radiographic data were collected from the records of 13 oral pathology laboratories. All cases were microscopically reviewed, considering the current World Health Organization classification of tumors and were classified according to histopathologic features. RESULTS: There were 43 females and 19 males (average age 33.9 years; range 8-63 years). Clinically, COdF lesions appeared as asymptomatic swellings, occurring similarly in the maxilla (n = 33) and the mandible (n = 29); 9 cases exhibited palatal depression. Imaging revealed well-defined, interradicular unilocular (n = 27), and multilocular (n = 12) radiolucencies, with displacement of contiguous teeth (55%) and root resorption (46.4%). Microscopically, classic features of epithelial-rich (n = 33), amyloid (n = 10), associated giant cell lesion (n = 7), ossifying (n = 6), epithelial-poor (n = 3), and granular cell (n = 3) variants were seen. Langerhans cells were highlighted by CD1a staining in 17 cases. Most patients underwent conservative surgical treatments, with 1 patient experiencing recurrence. CONCLUSIONS: To the best of our knowledge, this study represents the largest clinicopathologic study of COdF. Most cases appeared as locally aggressive lesions located in tooth-bearing areas in middle-aged women. Inactive-appearing odontogenic epithelium is usually observed within a fibrous/fibromyxoid stroma, occasionally exhibiting amyloid deposits, multinucleated giant cells, or granular cells.

Extraosseous odontogenic fibroma with a clear cells component: a 5-year follow-up.

The extraosseous/peripheral odontogenic fibroma (E/POF) is a benign mesenchymal odontogenic tumour found on the gingival surface with clinical characteristics identical to those of reactive lesions. A histopathological analysis is the only method for determining the difference between reactive lesions and these neoplastic lesions, whose recurrence potential varies between 38.9% and 50%, highlighting the importance of correct diagnosis. The following report describes an E/POF case with a clear cells component, as well as a long-term follow-up treatment, which we emphasise due to its potential for recurrence.

18F-FDG PET/CT in the Hyperparathyroidism-Jaw-Tumor Syndrome.

ABSTRACT: Hyperparathyroidism-jaw-tumor syndrome (HPT-JTS) is a rare autosomal dominant disorder. A typical manifestation of HPT-JTS is the association of jaw-ossifying fibroma with primary hyperparathyroidism. Due to its rarity and diversity in its manifestations, it is a challenging diagnosis. A 33-year-old woman was referred due to painful swelling of the right maxilla suggestive of malignancy. The clinical presentations were not conclusive until she underwent F18-fluorodeoxyglucose positron emission tomography/computed tomography (F18-FDG PET/CT). F18-FDG PET/CT proved to be a useful tool to assist the clinicians in visualizing the "bigger picture" and, therefore all manifestation as pieces of "one puzzle."

Desmoplastic fibroma of the Mandible with unusual histopathological features.

Desmoplastic fibroma (DF) is a rare myofibroblastic primary tumor of bone that histologically and biologically mimics the extra-abdominal desmoid tumor of soft tissue. The surgical management of this tumor has been a matter of controversy and its recurrence has been a matter of clinical relevance. In this case report, we describe an unusual case of DF in a 15-year-old male patient who presented with a slow-growing mass in the right posterior mandibular region of 2 years duration. The presence of areas mimicking Giant cell angiofibroma (GCA) is the highlight of the case. The possibility of misdiagnoses is more in early lesions since the available literature shows that immunohistochemistry (IHC) is not of much benefit while differentiating DFs from other spindle cell lesions.

Three-dimensional computed tomography cinematic rendering of mandibular odontogenic myxofibroma.

Three-dimensional (3D) methods for visualizing volumetric computed tomography (CT) data can aid in understanding the extent of a disease process and planning necessary surgical interventions. Recently, a new method of 3D image creation, known as cinematic rendering (CR), has been developed; it produces photorealistic images from standard CT acquisitions. We describe the CT appearance of a rare tumor type, odontogenic myxofibroma, in the mandible of a 33-year-old woman, and we focus on the potential applications of CR visualizations in this and similar scenarios. Although prospective data quantifying the advantages of CR relative to traditional methods of 3D visualization are still forthcoming, the photorealistic quality of CR images, nonetheless, suggests the important potential utility of this method. Realistic shadowing effects in the images create depth and show the relative positions of objects within a visualized volume in an advantageous manner. Furthermore, soft tissue details allow for visualization of structures that can otherwise be difficult to render with traditional methods.