A giant peripheral ossifying fibroma of the maxilla with extreme difficulty in clinical differentiation from malignancy: a case report and review of the literature.

BACKGROUND: Peripheral ossifying fibroma is a nonneoplastic inflammatory hyperplasia that originates in the periodontal ligament or periosteum in response to chronic mechanical irritation. Peripheral ossifying fibroma develops more commonly in young females as a solitary, slow-growing, exophytic nodular mass of the gingiva, no more than 2 cm in diameter. While various synonyms have been used to refer to peripheral ossifying fibroma, very similar names have also been applied to neoplastic diseases that are pathologically distinct from peripheral ossifying fibroma, causing considerable nomenclatural confusion. Herein, we report our experience with an unusual giant peripheral ossifying fibroma with a differential diagnostic challenge in distinguishing it from a malignancy. CASE PRESENTATION: A 68-year-old Japanese male was referred to our department with a suspected gingival malignancy presenting with an elastic hard, pedunculated, exophytic mass 60 mm in diameter in the right maxillary gingiva. In addition to computed tomography showing extensive bone destruction in the right maxillary alveolus, positron emission tomography with computed tomography revealed fluorodeoxyglucose hyperaccumulation in the gingival lesion. Although these clinical findings were highly suggestive of malignancy, repeated preoperative biopsies showed no evidence of malignancy. Since even intraoperative frozen histological examination revealed no malignancy, surgical resection was performed in the form of partial maxillectomy for benign disease, followed by thorough curettage of the surrounding granulation tissue and alveolar bone. Histologically, the excised mass consisted primarily of a fibrous component with sparse proliferation of atypical fibroblast-like cells, partly comprising ossification, leading to a final diagnosis of peripheral ossifying fibroma. No relapse was observed at the 10-month follow-up. CONCLUSIONS: The clinical presentation of giant peripheral ossifying fibromas can make the differential diagnosis from malignancy difficult. Proper diagnosis relies on recognition of the characteristic histopathology and identification of the underlying chronic mechanical stimuli, while successful treatment mandates complete excision of the lesion and optimization of oral hygiene. Complicated terminological issues associated with peripheral ossifying fibroma require appropriate interpretation and sufficient awareness of the disease names to avoid diagnostic confusion and provide optimal management.

Psammomatoid ossifying fibroma in the frontal sinus: An intriguing clinical encounter-A detailed case report.

BACKGROUND: Psammomatoid ossifying fibroma (POF) is a rare craniofacial neoplasm, primarily affecting the maxillofacial region, and typically observed in adolescents and young adults. This case report presents a unique occurrence of POF in a 50-year-old male, defying the conventional age range and exhibiting an unusual anatomical location within the frontal sinus. CASE: A 50-year-old male with a prior history of cecal adenocarcinoma and colectomy presented with left eye proptosis and new-onset headaches. Imaging revealed a well-defined calcified mass in the left frontal sinus, leading to a diagnosis of POF. Open surgical resection was performed to remove the tumor, and histopathological evaluation confirmed its diagnosis as psammomatoid ossifying fibroma. The patient exhibited no postoperative complications or signs of recurrence. CONCLUSION: This case underscores the diverse clinical presentations and diagnostic challenges associated with POF, emphasizing the importance of accurate diagnosis and multidisciplinary collaboration. Further research is needed to explore the genetic underpinnings and optimal management strategies for this intriguing condition.

Juvenile trabecular ossifying fibroma of the inferior turbinate.

Nasal obstruction is a commonly reported issue in the Otorhinolaryngology Outpatient Department. In this case, an early adolescent boy with a long-standing problem of right-sided nasal obstruction since childhood sought consultation. Diagnostic nasal endoscopy revealed a deviation of the nasal septum to the left, coupled with right inferior turbinate hypertrophy, all overlying healthy mucosa. A CT scan of the nose and paranasal sinuses further identified a bony hyperdense lesion with ground glass attenuation, confined to the right inferior turbinate. Subsequent biopsy confirmed juvenile trabecular ossifying fibroma (JTOF). The patient underwent endoscopic right medial maxillectomy, and the final histology affirmed the diagnosis of JTOF.

Ossifying fibroma mimiking jaw tumour: A radiographic dilema.

Fibro-osseous lesions (FOLs) of the craniomaxillofacial region comprise a group of developmental, dysplastic, and neoplastic alterations. FOLs include ossifying fibromas (OF), cemento-ossifying fibroma (COF), familial gigantiform cementoma (FGC), fibrous dysplasia (FD), and cemento-osseous dysplasia (COD). Evidence suggests that some FOL, especially FD and OF may have a risk of spontaneous malignant transformation. This report documents a rare case of malignant transformation of ossifying fibromas of the jaw and the probable cause for same. Although it is rare, the clinician should have a complete follow up to observe such changes among the patients having FOLs.

Imaging of Fibro-osseous Lesions and Other Bone Conditions of the Jaws.

This review directs the focus on the imaging features of various fibro-osseous lesions and other bone lesions that can be of similar presentation. Broad diagnosis of "fibrous osseous lesion" may culminate in improper treatment and management. Radiographic discriminating factors between these entities are highlighted and summarized to improve the diagnostic process when encountering these lesions.

Analysis of Diagnosis and Treatment Strategies for Primary Juvenile Psammomatoid Ossifying Fibroma Complicated With Primary Aneurysmal Bone Cyst.

BACKGROUND: Juvenile Psammomatoid Ossifying Fibroma (JPOF) is a type of noncancerous bone tumor that usually affects adolescents in the craniomaxillofacial area. Clinical manifestations are usually symptoms caused by the tumor's invasive compression of surrounding tissues. Aneurysmal Bone Cyst (ABC) is also a benign bone tumor, and it typically occurs in long bones and the spine. Only 2% to 3% of cases occur in the head and neck. Due to the rarity of this combination of clinical cases, clinicians face difficulties in comprehensively understanding this complex lesion. Therefore, a comprehensive review of the clinical manifestations and characteristic imaging findings is necessary for surgeons. CASE PRESENTATIONS: On April 6, 2019, a 13-year-old boy presented with left maxillofacial bulge and pain for 1 month. Magnetic resonance imaging of the paranasal sinuses showed an irregular hive-like mass signal in the left maxillary sinus, and cystic changes with fluid levels were seen in the lesion. After the initial diagnosis of JPOF with primary ABC, we decided to perform a facial mid-facial resection of maxillary sinus tumor to remove the tumor tissue. Finally, after 3 recurrences and 4 operations, there was no tumor recurrence for 20 months after the last operation, and the patient was still under continuous follow-up. CONCLUSIONS: This case provided a reference for the diagnosis and treatment of JPOF combined with ABC. In particular, a new understanding of the association between the two diseases and the management of recurrence were proposed, which had the potential to improve clinical understanding of this complicated condition.

Misdiagnosed long-standing unilateral nasal obstruction: ossifying fibroma of the inferior turbinate.

A man in his 20s presented with complaints of unilateral nasal obstruction for the past 6 years that progressively worsened leading to irrational use of over-the-counter nasal decongestants. With the worsening of symptoms, a non-contrast CT was done. It showed a dense expansile sclerotic lesion of the right inferior turbinate, which was excised endoscopically. Cemento-ossifying fibromas of the inferior turbinate are rare and require assessment and surgical excision to relieve the symptom of nasal obstruction. It derives its name from the variable proportions of fibrous and mineralised tissue present in it and exclusively develops in the craniofacial region. It can be surgically managed by an endoscopic, an endonasal non-endoscopic (with a speculum) or an open approach (lateral rhinotomy, sublabial approach or mid-facial degloving). Here, we present how such a case was detected and managed surgically by the endoscopic approach, which is a minimally invasive option with shorter hospital stay and early recovery.

Cemento-ossifying fibroma transforming to osteosarcoma.

Ossifying fibroma is a type of fibro-osseous lesion categorised into cemento-ossifying fibroma and juvenile ossifying fibroma. Malignant transformation of fibro-osseous lesions is documented especially for fibrous dysplasia, but scarcity is seen when we search for malignant transformation of ossifying fibroma. Thus, we are presenting an extremely rare case of cemento-ossifying fibroma transforming into osteosarcoma with long sequential radiographic details.

Giant fronto-spheno-orbitary juvenile psammomatoid ossifying fibroma: Case report and literature review.

Juvenile psammomatoid ossifying fibroma (JPOF) is an osteofibrous neoplasm that originates in the craniofacial skeleton typically during the first three decades of life. JPOFs usually involve the orbit, paranasal sinuses or the jaws. Extensive involvement of the anterior cranial base with compromised visual function is a rare phenomenon. In such clinical context, a definite diagnosis can only be made on the basis of histopathological findings, given the absence of pathognomonic radiological features. Despite being considered a benign entity, JPOFs present a locally aggressive behavior. Therefore, these neoplasms must be included in the differential diagnosis in every patient harboring a skull base osteofibrous lesion, and, once diagnosed, gross total surgical removal should be attempted. In this study, we present our experience in the diagnosis and treatment of a patient diagnosed with a giant JPOF involving the cranial base.

Benign bony lesions of paranasal sinuses and skull base: from osteoma to fibrous dysplasia.

PURPOSE OF REVIEW: Benign bony lesions of the craniofacial complex are relatively common. However, their location close to critical neurovascular structures may render their treatment, if required, highly challenging.This article reviews the current literature on their pathophysiology, diagnosis, natural course and treatment, with a focus on most recent findings. RECENT FINDINGS: A new classification has been suggested concerning endoscopic resectability. The ratio of lateral frontal to interorbital distance can accurately and reliably predict the endoscopic reach to lateral frontal sinus, while orbital transposition can assist us in reaching lateral frontal sinus when anatomy is unfavorable. New and combined endoscopic transnasal and transorbital approaches are now in the surgical armamentarium. Prophylactic optic nerve decompression in fibrous dysplasia is absolutely contraindicated as it leads to worse visual outcomes. Radiotherapy of such lesions is of no benefit and may lead to a higher risk of malignant transformation. The presence of Guanine Nucleotide binding protein Alpha Stimulating (GNAS) mutation in chromosome 20 is universally present in fibrous dysplasia and can differentiate them from ossifying fibromas. SUMMARY: Diagnosis and therapeutic management of benign craniofacial bone lesions remains challenging. If surgical treatment is contemplated, the morbidity of the intervention should always be weighed against the potential benefits. Evolution of extended endoscopic endonasal and transorbital surgery means that more lesions can be reached purely endoscopically with better oncological and cosmetic results.

Ossifying fibroma presented as an incidental [18F]FDG PET/CT uptake within the hard palate.

Incidental uptake in [18F]FDG PET/CT is not uncommon, but uptake in the oral and sinonasal regions was less frequently reported. We present a case of incidental focal [18F]FDG PET/CT uptake within the hard palate, which was later revealed to be an ossifying fibroma. We also reviewed some relevant literature and suggested that further investigation may be necessary for some patients with incidental [18F]FDG PET/CT uptake in the oral and sinonasal regions.

Computed tomographic characteristics of frontal sinus ossifying fibroma in a dog.

A 10-year-old female spayed Kelpie cross was presented to The Austin Vet Specialists for further investigation of a mineralized, lobulated frontal sinus mass that had previously been detected radiographically. Computed tomography (CT) revealed a large, expansile, well-defined, heterogeneously mineral attenuating mass invading both frontal sinuses. The mass was surgically debulked via a frontal sinusotomy approach. Histopathology was consistent with ossifying fibroma. This  is the first published report to describe frontal sinus ossifying fibroma in a dog, and the second to describe CT features of ossifying fibroma involving the cranium in a dog.

Spontaneous Regression of Ossifying Fibroma in the Oral and Maxillofacial Region: A Rare Case With 4-Year Follow-Up.

The case presented in this study is a rare instance of an ossifying fibroma (OF), a type of benign fibro-osseous lesions, spontaneously regressed without surgical intervention. A 9-year-old boy with an intraosseous lesion in the left maxilla was diagnosed as OF. The surgeon suggested surgical excision, but due to personal reasons, the patient and his parents deferred the surgery and opted for regular follow-up. During a 4-year follow-up, the surgeon found that the lesion had significantly decreased in size and the facial deformity had remitted. At the latest follow-up, the deformity of his left face became almost unnoticeable, and the lesion seemed to be subtle on cone-beam computed tomography images. This case highlights the possibility of spontaneous regression of OF. This phenomenon may occur due to the teeth eruption, which can lead to the formation of periodontal ligaments and engage the regression of OF.

Lesión Fibro-ósea de Mandíbula: Un Hallazgo Radiográfico / Fibro-osseous Lesion of the Mandible: A Radiographic Finding

Las lesiones fibro-óseas son consideradas benignas y componen un grupo de patologías de desórdenes que se caracterizan por el reemplazo de un hueso normal por un tejido compuesto de fibras colágenas, fibroblastos y tejido mineralizado. Presentamos un hallazgo radiográfico obtenido de un paciente de sexo masculino de 41 años de edad que asiste a un centro de radiología para realizarse una radiografía panorámica, el examen revela un interesante hallazgo radiográfico en la hemi mandíbula izquierda, donde se observa una lesión fibro-ósea con expansión ósea a nivel de reborde marginal y cortical basal mandibular, desplazamiento de canal mandibular, desplazamiento dentario, compromiso de cortical alveolar y rizálisis en diferentes niveles en los dientes adyacentes a la lesión. De acuerdo a los antecedentes anteriores se establece una hipótesis diagnóstica de Fibroma Osificante de larga data debido a su radiopacidad. La Organización Mundial de la Salud lo clasifica como una neoplasia ósea benigna con afección al esqueleto craneofacial, de mayor incidencia en mandíbula, se presenta generalmente entre la 3º y 4º década de vida. Concluimos que las lesiones fibro-óseas pueden ser detectadas como un hallazgo radiográfico, esto es relevante para un tratamiento precoz, sin embargo, el diagnóstico debe realizarse complementando los antecedentes clínicos e histopatológicos de la lesión, poniendo especial atención en el diagnóstico diferencial.

Fibro-osseous lesions are considered benign and make up a group of disorder pathologies that are characterized by the replacement of normal bone by tissue composed of collagen fibers, fibroblasts, and mineralized tissue. We present a radiographic finding obtained from a 41-year-old male patient who attended a radiology center for a panoramic radiograph. The examination revealed an interesting radiographic finding in the left hemi-mandible, where a fibro-osseous lesion was observed. with bone expansion at the level of the marginal ridge and basal mandibular cortex, displacement of the mandibular canal, dental displacement, compromise of the alveolar cortex and rizalysis at different levels in the teeth adjacent to the lesion. According to the previous antecedents, a long-standing diagnostic hypothesis of Ossifying Fibroma is established due to its radiopacity. The World Health Organization classifies it as a benign bone neoplasm affecting the craniofacial skeleton, with the highest incidence in the jaw, generally presenting between the 3rd and 4th decade of life. We conclude that fibro-osseous lesions can be detected as a radiographic finding, this is relevant for early treatment, however the diagnosis must be made by complementing the clinical and histopathological history of the lesion, paying special attention to the differential diagnosis.

Rapport de cas Ossifying fibroma in the cervical vertebra of a dog.

The objective of this study was to describe the clinical signs, neurologic examination findings, diagnostic imaging results, and pathologic diagnosis of ossifying fibroma in the cervical vertebra of a dog. A 3-year-old spayed female Pembroke Welsh corgi dog exhibited severe cervical pain and left-sided postural reaction deficits. Magnetic resonance imaging (MRI) revealed a lobulated contrast enhancing mass associated with the C6 cervical vertebra. Due to the lack of response to pain medications, humane euthanasia was elected, and histopathologic evaluation of the mass revealed a fibro-osseous lesion most consistent with an ossifying fibroma. This neoplasm is most commonly associated with the mandible of young horses and has not been previously reported in vertebrae in veterinary medicine. Key clinical message: This case is the first report of a fibro-osseous lesion most consistent with an ossifying fibroma affecting a vertebra in veterinary medicine.

Fibrome ossifiant dans la vertèbre cervicale d'un chien. Décrire les signes cliniques, les résultats de l'examen neurologique, les résultats de l'imagerie diagnostique et le diagnostic pathologique du fibrome ossifiant dans la vertèbre cervicale d'un chien. Une chienne Pembroke Welsh corgi femelle stérilisée âgée de 3 ans présentait de fortes douleurs cervicales et des déficits de réaction posturale du côté gauche. L'imagerie par résonance magnétique (IRM) a révélé une masse lobulée augmentant le contraste associée à la vertèbre cervicale C6. En raison de l'absence de réponse aux analgésiques, l'euthanasie a été choisie et l'évaluation histopathologique de la masse a révélé une lésion fibro-osseuse plus compatible avec un fibrome ossifiant. Ce néoplasme est le plus souvent associé à la mandibule des jeunes chevaux et n'a jamais été signalé auparavant dans les vertèbres en médecine vétérinaire.Message clinique clé :Ce cas est le premier rapport d'une lésion fibro-osseuse plus compatible avec un fibrome ossifiant touchant une vertèbre en médecine vétérinaire.(Traduit par Dr Serge Messier).

Juvenile psammomatoid ossifying fibroma of the maxilla and mandible: A systematic review of published case reports.

OBJECTIVE: The aim of this study is to evaluate recent evidence-based data that summarize the clinicopathological findings and treatment along with follow-up measures taken in terms of published cases of Juvenile psammomatoid ossifying fibroma (JPOF) of the maxilla and mandible by a systematic review. MATERIALS AND METHODS: The databases searched were PubMed, MEDLINE, Scopus, Google scholar, and Cross references. Only those case reports of JPOFs published in the English language from 2000 to 2022 were considered. All cases included confirmed JPOF lesions histopathologically. The SR-included details like clinical and radiographic data, follow-up details such as recurrence, and the presence of any adverse outcome. RESULTS: The database search produced 595 articles from 2000 to 2022, among which 22 case reports were included in the systematic review. The mean age of JPOF occurrence in patients was 18 ± 16 years. A male predilection was noted among patients younger than 14 years of age, whereas a female predilection was noted in patients older than 14 years of age. Frequent involvement of the mandible (56%) compared to the maxilla (44%) was reported. The posterior mandible was the most commonly affected site involving numerous adjacent structures. The expansile nature of the JPOF displayed 57% buccolingual expansion, 50% downward displacement or erosion of the lower border of the mandible and 81% of  involvement of the maxillary antrum/pterygoid plate/orbital floor. Among the 20 cases reported, the treatment provided included surgical excision in 45% of the patients, jaw resection in 35% of the patients, and enucleation and curettage in 18% of the patients. Follow-up details were provided in 80% of the reports that showed recurrence. CONCLUSIONS: The diagnosis of JPOF requires correlation of the clinical and radiographic features with key histopathological features. Although long-term follow-up of the case reports has been reported, the data lack information about the long-term outcomes of JPOF.

Ossifying Fibroma of Mandible - A Case Report.

INTRODUCTION: Ossifying fibromas are rare, non-aggressive benign tumours of the bone, commonly involving the posterior mandible in middle-aged individuals with a female predilection. FINDINGS: Clinical manifestations include asymptomatic expansion of the mandible with infrequent maxillary lesions, pain, malocclusion, and compromised quality of life including aesthetic perception. Owing to multiplicity of features, tendency of recurrence, and possibility of malignant transformation, the diagnosis, treatment, and post-operative management of ossifying fibroma are always a challenge. TAKEAWAY LESSONS: Study aims to report a clinical case of extensive swelling involving the coronoid process and condyle on the right side to crossing the mid-line of the mandible with compromised functions and aesthetics. The article describes the clinical, histopathological, and radiological features of the case. The possible treatment and challenges encountered are discussed.

Peripheral cemento-ossifying fibroma - A rare case report.

Peripheral cemento-ossifying fibroma (PCOF) is a relatively rare tumor classified between fibro-osseous lesions. PCOF is an osteogenic neoplasm that ordinarily presents as an epulis-like growth with uncertain pathogenesis. It predominantly affects young adults, with peak prevalence between the first and second decades of life. We present here the case report of a 15-year-old girl, which was previously surgically excised twice and had recurred both the times. The disease duration was 2 years and a follow-up of 1-year was done. Clinical, radiographic, and histological characteristics are discussed with recommendations regarding the treatment is provided.