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.

Case of metastatic clear cell odontogenic carcinoma with response to chemoimmunotherapy.

Our patient initially presented with 6 months of left jaw pain and gingival bleeding, leading to the discovery of a radiolucent left maxillary mass on dental evaluation. A biopsy confirmed clear cell odontogenic carcinoma, and the patient was treated with definitive surgery and radiation for localised disease. Unfortunately, the patient was found to have pulmonary metastases 3 months after initial management and was subsequently treated with a combination of cytotoxic chemotherapy and immunotherapy with a partial response. To our knowledge, this is the first case demonstrating the successful use of chemoimmunotherapy in metastatic clear cell odontogenic carcinoma.

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.

A Variant of Gorlin-Goltz Syndrome with Synchronous Malignant and Multiple Benign Lesions of the Jaws - A Case Report.

INTRODUCTION: Although numerous syndromic and non-syndromic odontogenic lesions of the jaws have been documented in the literature, there are very few cases of simultaneous benign and malignant jaw lesions. PATIENT CONCERNS: We present a case of right maxillary squamous cell carcinoma along with several benign odontogenic cystic lesions of the jaws and skeletal abnormalities that meet the criteria for Gorlin-Goltz syndrome. TAKEAWAY LESSONS: With a review of the literature, the specifics of management and follow-up are discussed.

Computed tomographic imaging features of maxillary and mandibular melanotic neuroectodermal tumor of infancy: Report of 2 cases and systematic review.

OBJECTIVE: The aim of this systematic review was to determine the computed tomographic (CT) imaging characteristics of maxillary and mandibular melanotic neuroectodermal tumor of infancy (MNTI). Two cases from our institution were also presented. STUDY DESIGN: Full-text case reports and case series of histopathologically proven gnathic MNTI with CT figures of diagnostic quality were searched in PubMed, Scopus, Web of Science, Ovid, and Google Scholar databases from July 2021 to February 2022. Descriptive statistics were used to determine the frequency of each CT feature of gnathic MNTI. RESULTS: Fifty-two published studies met the eligibility criteria, providing a total of 53 maxillary and mandibular MNTIs for analysis. In order of frequency, the CT features of gnathic MNTI that were present in over half of the study sample were bone expansion (53, 100%), a well-defined periphery (49, 92.5%), tooth displacement (45, 84.9%), and a bilocular radiolucent internal pattern (32, 60.4%). CONCLUSIONS: The bilocular radiolucent internal pattern has not been recognized as a common CT feature of gnathic MNTI. When associated with a well-defined, expansile mass in the infantile maxilla or mandible, this imaging characteristic can support a radiologic interpretation of MNTI.

Clinico-Pathological Characteristics, Management, and Outcome of Benign Intraosseous Maxillary Tumors in Adolescents: A Prospective Study.

INTRODUCTION: Benign intraosseous maxillary tumors, although uncommon among the adolescents, represent a major challenge to the maxillofacial surgeons. Treatment ranges from conservative surgery to radical resection and reconstruction. Maxillary defect reconstruction in adolescents is uncommon. it can be achieved by prosthetic obturators, local and regional flaps, and free-tissue transfer. AIM OF THE WORK: To investigate the clinical and pathological patterns, treatment, and outcome of benign intraosseous maxillary tumors in adolescents. PATIENTS AND METHODS: A 10-years prospective study included patients between 12-18 years who presented with benign intraosseous maxillary tumors and surgically treated (conservative surgery or radical resection) with immediate reconstruction. Cases were followed up to assess functional and aesthetic outcomes and detect complications. RESULTS: Study included 38 patients; 63.2% had non-odontogenic tumors and 36.8% had odontogenic tumors. The most common non-odontogenic tumor was central giant cell granuloma (31.6%). The most common odontogenic tumors were adenomatoid odontogenic tumor and ameloblastoma (10.5% each). Treatment included Conservative surgery (55.3%) and radical resection (44.7%). Reconstruction was performed in 17 cases by temporalis muscle flap (9.83%), obturator (2.46%), and free fibula flap (1.64%). All cases gained accepted functional and esthetic results. CONCLUSIONS: Non-odontogenic tumors were more common than odontogenic tumors. Central giant cell granulomas and ossifying fibromas were the most common non-odontogenic tumors. Ade-nomatoid odontogenic tumor and ameloblastoma were the most common odontogenic tumors. Maxillary reconstructions with tem-poralis muscle flap and obturator were simple and satisfactory. Microsurgical reconstruction had high success rates. Aesthetic, and functional results were satisfactory when appropriate reconstruction was performed.

Ameloblastic Fibro-Odontoma: At the Crossroad Between "Developing Odontoma" and True Odontogenic Tumour.

Ameloblastic fibro-odontoma (AFO) is a controversial, rare benign mixed odontogenic tumour that was re-defined as "developing odontoma" in the 2017 WHO classification arguing that once dental hard tissues form, it is programmed to transform into odontoma. However, AFO still remains unclear in terms of its nature. We aimed to analyze a large series of AFOs and compare it to a large series of odontomas (ODs) in an attempt to set cut-off diagnostic parameters between these entities and discuss latest updates on AFO histopathologic, clinical and molecular features. A total of 23 well-documented AFOs were analyzed versus 310 ODs focusing on the age of the patients and size of the lesions. For AFO, mean age was 9.4 ± 3.9 years (range 3-16 years) and mean size (greatest diameter) was 2.9 ± 1.5 cm (range 0.8-5.5 cm). For OD-mean age was 26.5 ± 15.6 years (range 3-81 years), mean size 1.9 ± 0.9 cm (range 1-5 cm). Receiver operating curve (ROC) showed that a cut-off age of 13.5 years and below [area under the curve (AUC) 0.902, 95%CI 0.859-0.945; p < 001; sensitivity 80%, specificity 87%] and a cut-off size of 2.1 cm and above are likely to be associated with AFO (AUC 0.7, 95%CI 0.574-0.827; p = 0.001; sensitivity 57%, specificity 77%). Thus, the combination of age and lesion size may be used to distinguish between lesions of a true neoplastic nature (i.e., AFO) and hamartomatous formation (i.e., OD). Further molecular and genetic specifications are needed to provide a better understanding on the pathogenesis of AFO in support of our suggestion and aid in an accurate classification of AFO.

Ghost cell odontogenic carcinoma of the left maxilla.

We report the case of a 25-year-old man with a maxillary ghost cell odontogenic carcinoma (GCOC). The patient presented to the maxillofacial and head and neck surgery clinic with a growing lump in the left maxilla. Initial workup with CT revealed a cystic lesion in the left upper jaw with associated bone erosion and an enhancing soft-tissue component. Enucleation showed a GCOC associated with a calcifying odontogenic cyst. After the diagnosis was obtained, the patient underwent widening of the first surgical resection. GCOCs are rare odontogenic neoplasms with unspecific clinical and imaging presentation, whose definitive characterisation is based on pathology. Current treatment approaches mainly involve surgical excision, but the prognosis is highly unpredictable due to intertumoral heterogeneity. As tumour recurrences occur in 73% of cases, radical surgery with negative margins is highly recommended. Therefore, it is essential to recognise this entity to offer patients adequate management.

Maxillectomy Reconstruction Revision Using Virtual Surgical Planning and Intraoperative Navigation.

Secondary revision of osseous flap reconstructions of the maxilla can enhance facial symmetry, but can be challenging due to the absence of normal anatomy and landmarks. We report four cases of maxillectomy reconstruction with scapula tip flap employing a novel combined approach with preoperative virtual surgical planning (VSP) and intraoperative navigation (ION) for secondary revision. VSP was employed to superimpose mirrored normal anatomy upon the reconstructed anatomy, and ION used for real-time intraoperative anatomical mapping. VSP and ION can be used to optimize maxillary bony revisions and recontouring, thereby improving anatomic symmetry and funtionality. Laryngoscope, 131:E2655-E2659, 2021.

Navigation-Assisted Resection and Fibula Free-Flap Reconstruction of an Extensive Maxillary Tumor.

OBJECTIVE: Tumor resection and reconstruction as a one-step procedure requires accurate definition of the intended safety margins, precise location of osteotomy lines and reliable individual rehabilitation.In recent years, the role of image-guided surgery in the maxillofacial region has increased significantly. As this technology allows the surgeon to track the actual position of each instrument during the operation in real-time, it makes it possible to perform extensive bone structure resections and reconstructions in anatomically distorted or complex areas, such as the head and neck region, without unnecessarily damaging vital structures.The authors described a case of a 26-year-old woman presented to our Clinic with an extensive maxillary squamous cell carcinoma involving the infratemporal fossa. The patient underwent single-step navigation-guided en bloc resection of the tumor and defect reconstruction.The aim of this study is describe and discuss the unusual surgical planning and the challenging operative technique adding a new case to the currently limited scientific literature on the computer-assisted head and neck oncologic surgery.

Vascularized Composite Parietal Bone Flap for Immediate Reconstruction of a Hemi-Maxillectomy Defect in an Infant.

BACKGROUND: Surgical resection of maxillary tumors can result in defects that can be difficult to reconstruct by conventional means due to the complex functional and anatomic nature of the midface and lack of regional bone flap options in the head and neck. Many reconstructive methods have been used to repair maxillary defects, but the ideal technique for the reconstruction of hemi-maxillectomy defects in growing pediatric patients has yet to be determined. METHODS: The authors present a rare pediatric patient with melanotic neuroectodermal tumor of infancy resulting in a hemi-maxillectomy defect after resection that was reconstructed using a pedicled vascularized composite flap consisting of temporalis muscle, pericranium, and parietal bone. RESULTS: The patient achieved successful long-term bony reconstruction of his right maxilla with this flap. Stable skeletal fixation with adequate orbital support was maintained over a >3-year follow-up period. CONCLUSION: A vascularized composite parietal bone flap is a reliable reconstructive option for reconstruction of large maxillectomy defects providing low donor-site morbidity, adequate globe support, excellent long-term skeletal stability, and malar symmetry in rapidly growing pediatric patients. Successful reconstruction for a rare patient with maxillary melanotic neuroectodermal tumor of infancy requiring hemi-maxillectomy was demonstrated with >3-year follow-up.

Do predetermined surgical margins compromise oncological safety in computer-assisted head and neck reconstruction?

OBJECTIVES: Computer assisted head and neck reconstruction has gained popularity over the past few years. In computer assisted surgery (CAS), surgical margins are predetermined in virtual surgery and resection guides are designed to be fitted intra-operatively. However, concerns have been raised regarding the oncological safety of predetermined surgical margins. Therefore, the aim of this study was to compare surgical margins, recurrence and survival outcomes in patients underwent CAS and non-CAS in head and neck reconstruction. METHODS: We retrospectively reviewed the patients underwent oral and maxillofacial malignancies surgical excision and free flap reconstruction from October 2014 to December 2019 by the same chief surgeon. Patients were divided into two groups depending on whether CAS and predetermined surgical margins were adopted. The primary outcome was surgical resection margin and the secondary outcomes included recurrence and survival. RESULTS: A total of 66 subjects were recruited with 37 in the CAS group and 29 in the non-CAS group. The follow-up rate was 100%. The average follow-up time was 24.5 months. No significant difference in resection margin was identified between the groups (p = 0.387). Tumor staging, margin status, perineural invasion, lymphovascular invasion and extranodal extension were identified as significant factors influencing survival. Both before and after adjustment for these prognostic factors identified, CAS and non-CAS group showed no significant difference in survival outcome. CONCLUSION: Predetermined surgical margins do not compromise oncological safety in terms of resection margin, disease recurrence and patient survival.

Surgical Treatment of Aggressive Intraosseous Cavernous Hemangioma in Maxilla Through Surgical Resection.

Hemangiomas are benign vascular lesions characterized by endothelial vascular proliferation and may demonstrate aggressive clinical features. Intraosseous hemangiomas are uncommon and the maxillary location rare, with few cases described in the literature. The treatment of these lesions varies, however they have a better prognosis when a surgical treatment through resection is performed. The aim of this study is to report a case of right maxillary cavernous intraosseous hemangioma treated by surgical resection with previous embolization. The diagnosis was made through incisional biopsy and immunohistochemical examination. An aggressive lesion profile was observed through the Ki67 marker. During surgery, a temporary ipsilateral external carotid artery ligation was performed to minimize possible bleeding. Weber-Fergson surgical access was performed and total surgical resection through hemi-maxillectomy. The patient is stable after 30 months of follow-up without relapses. In suspected cases of intraosseous hemangiomas, accurate diagnosis and total surgical resection of the lesion is essential. The use of therapeutic mammoths that minimize sagging during and after the procedure such as embolization and arteriography should always be performed.

Case series of adenomatoid odontogenic tumour of jaws with variations in presentation.

Adenomatoid odontogenic tumour (AOT) is a rare tumour of odontogenic origin with distinct clinicopathological appearance but is often clinically misdiagnosed as a cyst. The most common site is the anterior maxilla in the canine region. We present here two cases, one at its commonest location in the maxillary canine while the other is at the uncommon location of the anterior mandible. Its clinical features of painless slow growing swelling, association with impacted tooth and radiographic appearance of well defined predominantly radiolucent lesion are overlapping with other oral pathologies like dentigerous cyst, radicular cyst, calcifying odontogenic cyst, calcifying epithelial odontogenic tumour, unicystic ameloblastoma etc. One must be aware and updated with the variation in appearance of AOT. Encapsulation of tumour causes less cumbersome enucleation of the tumour, a successful treatment as it reduces the chances of recurrence.

Squamous cell carcinoma at sites of old maxillary fractures.

Malignancies have been reported to occasionally arise in scar tissue following injury. One hypothesis involves prolonged overactivation of tissue repair systems due to chronic inflammation and irritation, although the pathogenesis of cancers occurring in scars is not fully understood. We describe here two cases with a history of maxillary fracture at the site where squamous cell carcinoma (SCC) subsequently developed. The first patient developed SCC 7 years after right maxillary fractures resulting from a traffic accident. He underwent chemoradiotherapy (70 Gy in 35 fractions) and maintained complete response (CR) for 10 months. The second patient developed SCC 3 years after sustaining right maxillary fractures in an ice hockey game. Radiotherapy and total maxillectomy were performed, but local recurrence arose and he has since been receiving chemotherapy.