Odontogenic carcinoma with dentinoid: case report and literature review of a rare entity.

BACKGROUND: Odontogenic carcinoma with dentinoid (OCD) is a rare and controversial entity, which has not yet been included in the current World Health Organization classification of odontogenic lesions. Owing to the small number of reported cases, the clinicopathological characteristics, biological behavior, prognosis, and appropriate treatment strategies for OCD remain to be defined. Herein, we present an additional case of OCD with a focus on the differential diagnosis and review of the pertinent literature, in order to enable better recognition by oral clinicians and pathologists and further characterization of this entity. CASE PRESENTATION: This paper reports a case of OCD in the posterior mandible of a 22-year-old female. Radiography showed a well-defined unilocular radiolucency with radiopaque materials. The intraoperative frozen section pathology gave a non-committed diagnosis of odontogenic neoplasm with uncertain malignant potential. Then a partial mandibulectomy with free iliac crest bone graft and titanium implants was performed. Microscopically, the tumor consisted of sheets, islands, and cords of round to polygonal epithelial cells associated with an abundant dentinoid matrix. Immunohistochemically, the tumor cells were diffusely positive for CK19, p63, and ß-catenin (cytoplasmic and nuclear). No rearrangement of the EWSR1 gene was detected. The final diagnosis was OCD. There has been no evidence of recurrence or metastasis for 58 months after surgery. We also provide a literature review of OCD cases, including one case previously reported as ghost cell odontogenic carcinoma from our hospital. CONCLUSIONS: OCD is a locally aggressive low grade malignancy without apparent metastatic potential. Wide surgical excision with clear margins and long-term period follow-up to identify any possible recurrence or metastases are recommended. Histopathological examination is essential to conclude the diagnosis. Special care must be taken to distinguish OCD from ghost cell odontogenic carcinoma and clear cell odontogenic carcinoma, as misdiagnosis might lead to unnecessary overtreatment. Study of additional cases is required to further characterize the clinicopathological features and clarify the nosologic status and biological behavior of this tumor.

Central giant cell granuloma in the posterior region of mandible mimicking a fibro-osseous lesion and hemangioma: a case report.

BACKGROUND: A central giant cell granuloma (CGCG) is a benign, proliferative, intraosseous, and non-odontogenic lesion occurring primarily in children and young adults. On the histological level, it is characterized by numerous multinucleated giant cells scattered randomly throughout a sea of spindle-shaped mesenchymal stromal cells which are dispersed throughout the fibrovascular connective tissue stroma containing areas of haemorrhage. When it comes to radiographic features, CGCG can have an array of variations, ranging from well-defined expansile lesions to ill-defined and destructive lesions, with or without expansion. CASE PRESENTATION: This case report reviews an 11-year-old Caucasian patient with a chief complaint of slow-growing swelling involving the right posterior mandibular region. The cone beam computed tomography (CBCT) revealed an ill-defined mixed lesion mimicking both fibro-osseous lesion and hemangioma. However, microscopic examination revealed multinucleated giant cells in a fibrous stroma suggestive of central giant cell granuloma. CONCLUSION: Our intent in reporting this case is to highlight the importance of thorough clinical, radiographical and histopathological examination for accurate diagnosis and therapeutic interventions as well as to emphasize the importance of taking different possibilities into consideration when examining bony swellings in the head and neck region.

Aggressive presentation of ameloblastic fibro-odontoma: a clinical-pathological enigma.

Ameloblastic fibro-odontoma (AFO) is a rare, gnathic, benign, mixed odontogenic tumor that commonly presents in the first or second decade of life as a unilocular and rarely multilocular radiolucency with variable amounts of calcified material. Tumor progression is typically indolent, and generally accepted treatment is surgical enucleation and curettage. This case report describes an atypical presentation in a 14-year-old male with a multilocular, aggressive AFO requiring hemimandibulectomy with immediate osseous and dental "Jaw-in-a-Day" reconstruction. This report highlights the debate regarding whether AFO is a true neoplasm or an early-stage hamartoma in the continuum of complex odontoma formation. Regardless of the pathogenesis, maxillofacial surgeons and pathologists should be cognizant of the potential for AFO to develop locally aggressive behavior with considerable morbidity.

Mandibular rhabdomyosarcoma with TFCP2 rearrangement and osteogenic differentiation: a case misdiagnosed as fibrous dysplasia or low-grade central osteosarcoma.

Rhabdomyosarcoma with TFCP2-related fusions (TFCP2-RMS) is a rare entity that commonly affects young adults with a predilection for skeletal involvement. We herein report a 40-year-old female patient with TFCP2-RMS who was misdiagnosed as fibrous dysplasia or low-grade central osteosarcoma of the mandible by referring institutions. Histologically, the tumor showed dominant spindle cells and focal epithelioid cells with marked immature woven bone formation. Immunophenotypically, in addition to the characteristic expression of myogenic markers, ALK, and cytokeratins, tumor cells also unusually expressed osteogenic markers, such as MDM2 and SATB2. Through fluorescence in situ hybridization, the tumor cells showed EWSR1::TFCP2 gene fusion and no MDM2 gene amplification. This is a rare case of TFCP2-RMS, which was misdiagnosed as low-grade central osteosarcoma due to its presenting immunophenotype of MDM2 and SATB2, as well as extensive osteoid matrix formation.

Clinicoradiologic features of ameloblastomas: A single-centre study of 155 cases.

BACKGROUND: The purpose of the current study was to report on the clinical presentation and radiologic features of 155 cases of ameloblastoma (AB), representing a detailed, large, single-centre radiologic study. METHODS: Histologically confirmed cases were reviewed over 11 years. Demographic and clinical data were retrieved from the patient's records. Radiologic information was analysed from available radiographs. The radiologic features of ABs were assessed according to the mean age of presentation and the mean duration of the lesion. The distinguishing radiologic features between adults/children and sex were also evaluated. RESULTS: A statistically significant correlation existed between loss of border demarcation and advanced mean age. Multilocular lesions were markedly more common in adults compared to children. Multilocular ABs were associated with increased lesion duration and advanced mean age. Radiologic signs of reactive bony changes associated with the tumour presented at the highest mean duration of all bony effects. Bony expansion and cortical destruction were statistically correlated with lesion duration. Tooth impaction was more common in children. Some mandibular lesions reached a significant size, resulting in impingement of the maxillary sinus, zygoma, orbit and pterygoid plates. CONCLUSION: Due to unfortunate healthcare access constraints, ABs grow to significant sizes and exhibit features not often reported in the literature. The findings of this analysis highlighted the radiologic features of ABs expressed through the mean age and duration of the lesion. This emphasises the significance of timely management of these lesions.

Intraosseous schwannoma of the mandible: new case series, literature update, and proposal of a classification.

Intraosseous schwannoma of the mandible is rare, with diagnostic and therapeutic challenges. The aims of this study were to report new cases of intraosseous schwannoma of the mandible and to propose a clinical classification, providing suggestions for treatment methods. The cases of 13 patients treated at the authors' hospital and 86 cases reported previously in the literature were reviewed. The most common clinical feature was facial swelling (60/93). The rate of cortical thinning or expansion was 44.8% (43/96); widening of the inferior alveolar nerve canal on radiographs was observed in 15 patients.

Influence of microvascular mandibular bony reconstruction on the posterior airway space: A retrospective cohort study.

INTRODUCTION: The posterior airway space (PAS) is a common site of passive obstructions with high morbidity. Surgical changes to the craniomandibular system may affect the PAS. Data regarding the effects of mandibular reconstruction using vascularized bone flaps on PAS are insufficient. This retrospective cohort study aimed to investigate changes in PAS after mandibular reconstruction. MATERIALS AND METHODS: Pre- and post-reconstructive computed tomography scans of 40 patients undergoing segmental mandibulectomy and mandibular reconstruction with deep circumflex iliac artery or fibula flaps were analyzed. Absolute differences in PAS geometry and relative trends of PAS volume changes were compared within the study population and between subgroups formed according to the extent of resection, timing and type of reconstruction, and presence of pre-reconstructive radiotherapy. RESULTS: Irradiated patients were characterized by an increase in PAS volume after reconstruction. Absolute differences in total PAS volume after reconstruction were significantly different (p = 0.024) compared to non-irradiated patients. Reconstruction of central mandible segments resulted in decrease of the cross-sectional PAS areas. Absolute differences in middle cross-sectional PAS area after reconstruction were significantly different (p = 0.039) compared to non-central reconstructions. Patients who received radiotherapy were less likely to show a total PAS volume reduction after reconstruction (OR: 0.147; p = 0.007), with values adjusted for gender, age, body mass index, timing and type of reconstruction, and transplant length. CONCLUSIONS: Mandibular reconstruction causes changes in PAS geometry. Specifically, reconstructions of central mandibular segments can lead to a reduction in the cross-sectional areas of PAS, and mandibular reconstructions in irradiated sites may cause an increase in PAS volume.

Clinicopathologic Profile and Surgical Modalities in Mandibular Ameloblastoma: A Descriptive Study.

BACKGROUND: Ameloblastoma is a benign neoplasm composed of epithelial tissue with invasive and infiltrative behavior at the local level and a high recurrence rate, with various histopathologic patterns and clinical forms. Approximately 85% of conventional ameloblastomas occur in the mandible, most often in the body, angle, and ascending ramus area. The treatment modalities include both conservative and radical treatments. Postoperative follow-up is most important in the treatment of ameloblastoma. AIMS AND OBJECTIVES: To describe the clinicopathologic profile of mandibular ameloblastoma in patients undergoing different surgical modalities. The primary objective was to describe the clinicopathologic profile and surgical management of mandibular ameloblastoma in patients aged ≥18 years, who had reported to a tertiary dental care center for follow-up during the study period. The secondary objective was to describe the distribution of comorbidities associated with different surgical modalities and reconstructive methods. SUBJECTS AND METHODS: A total of 34 patients with mandibular ameloblastoma who underwent various surgical modalities between 2011 and 2021 were studied. Information was collected using a predesigned proforma and statistically analyzed. RESULTS: Thirty-four review cases of ameloblastoma were included in the study. The patients were analyzed concerning age, sex, site, size, clinical presentation, radiographic pattern, histopathologic subtype, type of surgery, and associated comorbidities. Most cases of mandibular ameloblastoma involve the age of 16 to 55 years. The mean age of occurrence was found to be 35.5±13.2. A female preponderance, a tumor size range of 2 to 4 cm, a multicystic variant, involvement of the mandibular body in the premolar-molar area, root resorption, cortical perforation, and a follicular type of histopathologic pattern were the common presentations. Isolated anterior tumors restricted to the incisor/canine region were not found. The common surgical modalities undertaken were conservative methods such as enucleation, and chemical cauterization, and radical methods such as marginal mandibulectomy and segmental resection. Reconstruction using a titanium plate or free fibular graft was performed in the indicated cases. The common comorbidities included difficulty in chewing and loss of facial contour. Recurrence after surgical treatment was rare. Only 9% of cases developed a recurrence within 5 years. No recurrence was noted in cases treated with radical treatment, whereas 50% of cases treated with conservative methods showed recurrence. CONCLUSION: The age of occurrence, site, and size of the tumor, cortical perforation, root resorption, histopathologic type, and radiographic patterns are widely considered factors in devising a treatment plan for mandibular ameloblastoma. However, there may be rare instances where these tumors behave differently regardless of their innocuous clinicopathologic presentation. Surgical procedures such as segmental resection and marginal mandibulectomy were found to be promising for the eradication of the tumor, and prevention of recurrences and metastasis. However, conservative measures such as enucleation and chemical cauterization were fraught with an increased risk of tumor recurrence and metastasis. Future studies with a larger sample size should focus on the clinicopathologic characteristics of ameloblastoma to elucidate its varied behavior and develop newer and advanced treatment modalities that would provide better surgical and postsurgical outcomes in affected patients.

Virtual planning, guided surgery, and digital prosthodontics in the treatment of extended mandible chondrosarcoma.

Chondrosarcoma is among the most common primary bone tumors in adults. In the mandible, chondrosarcoma is a very uncommon malignant cartilage-producing tumor. This case report shows how virtual planning combined with other digital technologies may improve masticatory function rehabilitation in patients with enlarged mandibular chondrosarcoma. The present study reports a case of a 52-year-old male patient who was initially diagnosed with a mandible chondroma, which was successfully excised with no evidence of malignant transformation. Nevertheless, the patient's symptoms recurred after 10 years, and a subsequent diagnosis of mandible chondrosarcoma was established, prompting the need for subtotal mandible resection and reconstruction with a fibula-free flap. Following a healing period, the patient underwent dental implant surgery to restore the mandibular dental arch, which was performed utilizing computer-aided design and computer-aided manufacturing technology, with fully guided implant placement facilitated by virtual planning. In this case report, the implant position data merging process is described from the digital impression and control model to ensure optimal passive fit of the full-arch zirconia prosthesis and discuss the importance of occlusal adjustments to avoid technical and biological complications. Virtual planning and digital technologies are crucial for the effective management of mandibular defects, allowing for accurate treatment and complete restoration of mandibular function. Their use leads to improved patient outcomes and quality of life. As technology advances, their importance in treating complex medical conditions is only expected to grow.

Two pathological fractures due to mandibular metastasis, rare in colon cancer; a case report presentation.

We reported on 65 years old patient who has colon cancer and referred to our palliative care center with pain due to enlarging metastatic mass on the dorsal of the right hand. She had swelling and numbness on her jaw. Computed tomography (CT) scan was performed for mandible imaging and two pathologic fractures were detected on the right corpus and right condyle of the mandible. Clinicians should consider possible metastases for terminal stage cancer patients.

Ameloblastic fibro-dentinoma: a rare mixed odontogenic tumor case report with review of literature.

BACKGROUND: Ameloblastic fibro-dentinoma is considered a rare, benign, mixed odontogenic tumor that occurs mainly in the posterior mandible in the 1st-2nd decade of life. Although the clinical behavior of Ameloblastic fibro-dentinoma is similar to that of ameloblastic fibroma, there is a debate about whether Ameloblastic fibro-dentinoma is a developing hamartomatous odontoma or a separate neoplastic odontogenic tumor like ameloblastic fibroma. However, it is important to understand the histopathogenesis of this rare tumor. CASE PRESENTATION: A case report presenting an 11-year-old male child with a swelling in the posterior mandible. Radiographic examination revealed a multilocular lesion with mixed radiodensity related to the impacted lower left second premolar tooth. Incisional biopsy was done, and microscopic examination revealed cords and nests of odontogenic follicles lined by ameloblast-like cells and central stellate reticulum-like cells in the primitive ecto-mesenchymal stroma with areas of dentinoid material and osteodentin. The diagnosis was ameloblastic fibro-dentinoma. Surgical excision of the lesion was done, and the patient was followed up for 1 year without evidence of recurrence. CONCLUSION: Reporting such a rare entity clarifies the debate about its nature and the importance of early diagnosis of lesions that are associated with unerupted teeth showing how it is effective in early management and prognosis.

Impact of the adjacent bone on pseudarthrosis in mandibular reconstruction with fibula free flaps.

BACKGROUND: Mechanical and morphological factors have both been described to influence the rate of pseudarthrosis in mandibular reconstruction. By minimizing mechanical confounders, the present study aims to evaluate the impact of bone origin at the intersegmental gap on osseous union. METHODS: Patients were screened retrospectively for undergoing multi-segment fibula free flap reconstruction of the mandible including the anterior part of the mandible and osteosynthesis using patient-specific 3D-printed titanium reconstruction plates. Percentage changes in bone volume and width at the bone interface between the fibula/fibula and fibula/mandible at the anterior intersegmental gaps within the same patient were determined using cone-beam computed tomography (CBCT). Additionally, representative samples of the intersegmental zones were assessed histologically and using micro-computed tomography (µCT). RESULTS: The bone interface (p = 0.223) did not significantly impact the change in bone volume at the intersegmental gap. Radiotherapy (p < 0.001), time between CBCT scans (p = 0.006) and wound healing disorders (p = 0.005) were independent risk factors for osseous non-union. Preliminary analysis of the microstructure of the intersegmental bone did not indicate morphological differences between fibula-fibula and fibula-mandible intersegmental bones. CONCLUSIONS: The bone interface at the intersegmental gap in mandibular reconstruction did not influence long-term bone healing significantly. Mechanical and clinical properties seem to be more relevant for surgical success.

A case of sclerosing odontogenic carcinoma of the mandible with a review of the literature.

Sclerosing odontogenic carcinoma (SOC) is a very rare malignant odontogenic tumor characterized by sclerotic stroma and single-file cord-like tumor cell structures. A 38-year-old man presented with extraoral swelling and right mental region paralysis. Panoramic radiography revealed an ill-defined radiolucent lesion extending from the right mandibular ramus to the right lower canine. Magnetic resonance imaging showed tumor invasion into the right inferior alveolar nerve and masseter muscle. Hemimandibulectomy, bilateral neck dissection, and mandibular reconstruction were performed using a rectus abdominis musculocutaneous flap and a titanium plate. Histopathology and immunohistochemistry confirmed SOC diagnosis. No recurrence occurred in the 1-year follow-up. In this paper, a case of SOC with a high Ki-67 labeling index was reported. Since SOC is prone to nerve invasion, treatment is resection with an appropriate surgical margin.

An Oroauricular Fistula After Mandibular Osteosarcoma Resection.

Bone tumors are rare malignancies and osteosarcoma is the most common malignant bone tumor. However, only 6% of all osteosarcomas occur in the mandible and maxilla 1. The location of these tumors also presents a unique set of challenges during resection and repair when compared to long bone tumors.

Unicystic ameloblastoma - A case series.

Ameloblastoma is a benign, locally aggressive neoplasm that constitutes about 1-3% of the tumors of the jaw. Wide surgical excision with adequate safe margin is the most common treatment of choice. The study aimed to manage cases with unicystic ameloblastoma while preserving the continuity of the mandible (without resection). This article presents a series of cases ranging from 18 to 40 years old patients of both sexes with unicystic ameloblastoma, especially in the mandible showing more male predilection than female. All the cases presented in this article were treated by enucleation and curettage. None of the patients presented post-operative paresthesia. None of the cases went in for resection. Post-operative recovery was uneventful in all the patients. All the patients were followed up for a period of 3.5-5 years. None of the cases reported recurrence at the date of publication.

Mandibular reconstruction after excision of recurrent odontogenic keratocyst using a novel mandibular distraction osteogenesis method- a case report.

BACKGROUND: Odontogenic keratocyst is one of the most common benign odontogenic neoplasms with a high recurrence rate. Its resection has the potential to lead to mandibular segmental defects. In this case report, we describe a patient with odontogenic keratocyst who underwent radical resection using a novel distraction osteogenesis (DO) method to reconstruct mandibular segmental defect. CASE PRESENTATION: This case report describes a 19-year-old woman with odontogenic keratocyst of the mandible that recurred after multiple curettages and eventually necessitated radical resection. Mandibular segmental defect after radical resection was reconstructed using a novel DO method that involved directly contacting the segment ends of the defect without the transport disk. However, the distractor broke during the retention period, and a molding titanium plate was used for fixation. This novel distraction method achieved mandibular reconstruction and restored mandibular function and contour.

Solitary intraosseous myofibroma: a rare case diagnosed from mandibular fracture.

Intraosseous myofibroma is a rare tumor of benign nature, slow growth, and low morbidity. The aim of this article is to report a case of pathologic fracture associated with the incidental diagnosis of myofibroma in the mandible of an adolescent. A 15-year-old girl reported that she experienced a physical assault resulting in facial injuries 1 month previously and had since experienced severe pain, malocclusion, and chewing difficulty. The cone beam computed tomographic examination revealed multiple features suggestive of pathologic fracture associated with a hypodense lesion with lobulated limits, as well as expansion and thinning of the cortical bone in the left mandible. The histopathologic diagnosis of the lesion indicated myofibroma. Treatment consisted of enucleation and curettage of the lesion with reduction and internal fixation of the fracture. After 18 months, the osteosynthesis plates and an impacted mandibular third molar were removed. Curettage of the lesion in association with treatment of the mandibular fracture proved to be effective for both bone consolidation and absence of recurrence while restoring mandibular functionality.