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.

Oncologic and functional results between sentinel lymph node biopsy and elective neck dissection in cT1/2N0 maxillary squamous cell carcinoma.

OBJECTIVE: To evaluate the oncologic safety and quality of life associated with the use of sentinel lymph node biopsy (SLNB) as compared to elective neck dissection (END) in patients with cT1/2N0 maxillary squamous cell carcinoma. METHODS: This study constituted a retrospective analysis of consecutively treated patients who underwent SLNB or END, with data collected prospectively. We analyzed the impact of the different neck procedures on regional control and disease-specific survival via the Cox model. Patients in both groups completed the University of Washington Quality of Life questionnaire. RESULTS: We included a total of 130 patients, with 47 receiving SLNB. In all cases, the sentinel lymph node could be identified, and of these, 5 had a positive result, yielding a sensitivity of 83.3 %, a specificity of 100 %, a false negative rate of 16.7 %, and a negative predictive value of 97.6 %. The sensitivity, specificity, false negative rate, and negative predictive value of END in detecting occult metastasis were 64.3 %, 100 %, 35.7 %, and 93.2 %, respectively. In comparison to END after propensity score matching, SLNB exhibited no significant difference in its effects on regional control (p = 0.519, HR: 1.05, 95 % CI: 0.52-1.93) and disease-specific survival (p = 0.634, HR: 1.22, 95 % CI: 0.53-1.99). Patients in SLNB group showed significantly higher mean scores of shoulder and taste domains at 3 months, 6 months, and 12 months postoperatively compared to those in END group. CONCLUSION: SLNB could act as a viable alternative to END in cT1/2N0 maxillary squamous cell carcinoma with comparable prognosis and better quality of life.

Central odontogenic fibroma: report of 29 cases in a Korean population with tooth management.

This study presents the behavioural findings of central odontogenic fibroma (COF) in a specific ethnic group, analysing treatment methods and demonstrating how involved teeth should be managed in detail. Clinical, radiographic, and histological findings were gathered for 29 patients who visited our clinic, with all patients' data carefully examined by radiologists and reviewed microscopically. The cohort comprised 29 patients, with 16 females and 13 males, having a mean (SD) age of 33.1 (16.0) years. Among them, 19 patients were affected in the maxilla, with 15 showing anterior preference, and palatal depression was observed in six patients. Tooth resorption was evident in 15 patients, while 10 patients showed tooth displacement. Within the cohort, 13 patients underwent tooth extraction and resection, while the remaining 16 did not have teeth extracted. Notably, there was no significant difference in recurrence observed between these two groups. This study represents the largest study to date of COF within a single ethnic group and institution. A subset of cases exhibited noteworthy features of COF. However, intriguingly, despite these characteristics, the preservation of contiguous teeth did not demonstrate a significant impact on recurrence rates.

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.

An Unusual Maxillary Tumor with Tubuloductal Epithelial Structures, Solid Epithelial Nests and Stromal Odontogenic Ameloblast-Associated Protein Deposits. Tubuloductal/Syringoid Variant of Central Odontogenic Fibroma with Amyloid?

Glandular tumors of jaw bones present, most often, histopathologic features of salivary gland and, rarely, of cutaneous glandular neoplasms. They are thought to originate from odontogenic epithelium. An unusual maxillary tumor presenting as a radiolucency in the periapical area of the right permanent lateral incisor of a 74-year-old male is presented causing root resorption. Preparations revealed occasionally branching tubular cords and ductal structures characterized, mostly, by a bilayer composed of luminal cuboidal to low columnar cytokeratin (CK) 7, Ber-EP4 and occasionally CK8/18 positive cells, and abluminal, CK5/6 positive, basal/basaloid cells revealing nuclear reactivity for p63/p40. Smooth muscle actin and calponin were negative, save for a single focus of calponin positive cells, confirming absence of myoepithelial support or epithelial mesenchymal transition. CK19 exhibited staining of both layers, the luminal being more intense. Eosinophilic secretory material and, occasionally, a luminal pellicle were decorated with CK8/18 and polyclonal carcinoembryonic antigen (CEA). CD1a identified only rare Langerhans' cells and Ki67 decorated 1-2% of abluminal cell nuclei. Small solid nests of epithelial cells were also present. Infrequently, an apparent transition of a nest into a tubular structure was appreciated. The partially inflamed stroma featured multiple hyalinized acellular deposits consistent with amyloid, as confirmed by bright orange Congo red reactivity with apple-green birefringence, which reacted with odontogenic ameloblast-associated (ODAM) protein antibody but not with antibodies for amelotin and secretory calcium-binding phosphoprotein proline-glutamine rich 1. Based on the above, the diagnosis of tubuloductal/syringoid variant of central odontogenic fibroma with ODAM amyloid is favored.

The Zygomatic Implant Perforated (ZIP) flap reconstructive technique for the management of low-level maxillary malignancy - clinical & patient related outcomes on 35 consecutively treated patients.

BACKGROUND: The zygomatic implant perforated (ZIP) flap technique provides immediate reconstruction and rapid dental rehabilitation for low-level malignant tumors. METHODS: Patients who underwent ZIP flap reconstruction between December 2015 and February 2021 were followed prospectively. RESULTS: Thirty-five consecutively treated patients were studied with 16 undergoing surgery alone and 19 undergoing surgery followed by radiotherapy. The median time to fit the prosthesis was 29 days with all patients requiring adjuvant radiotherapy receiving their fixed dental prosthesis prior to its commencement. Vascularized flap (100%), zygomatic implant (98.4%), and prosthesis (97%) survival were excellent and the ZIP flap protocol was highly rated by patient-related outcome measures especially for the chewing domain. CONCLUSIONS: The ZIP flap technique provides an excellent means of providing an autogenous oronasal seal and a foundation for immediate cortically anchored fixed dental rehabilitation. CLINICAL SIGNIFICANCE: This technique provides rapid and robust rehabilitation for patients presenting with low-level maxillary malignancy despite the use of radiotherapy.

[Oral Function Restoration via Two-Stage Reconstructive Treatment of an Extensive Maxillary Defect Following Maxillectomy].

Recently, the number of survivors that had oral cancer has increased, but deterioration in the quality of life of patients concerning oral function and cosmetic appearance has become problematic. Prosthodontic dentures usefully treat jaw defects after maxillary resection for cancer, but advances in microsurgery have rendered the possibility to reconstruct maxillary defect and oral cavity using a microvascular flap. Here, we describe 2-stage treatment after 35 years postoperatively. We restored the maxillofacial function using microvascular flaps and partial denture. The patient was a 59-year-old woman who underwent left lateral maxillectomy 35 years previously to treat an advanced left maxilla gingival carcinoma. A maxillary prosthesis was applied, requiring repeated adjustments and refabrications. The patient visited to obtain improvements for oral and maxillofacial functions and long-term quality of life. The left maxillary bone defect was 45×50 mm in area. Collaborating with the Department of Plastic and Reconstructive Surgery, the patient underwent reconstructive treatment using a microvascular forearm flap and partial denture. The patient's progress was excellent; eating, swallowing, and articulation improved. For 1 year postoperatively, the patient was satisfied with the results.

[Surgical Treatment of Maxillary Gingival Carcinoma with Mental Retardation-A Case Report].

Many patients with mental retardation have a limited awareness of abnormalities in the oral cavity, making early detection of oral cancer difficult. Moreover, during examination and treatment, these patients may not understand well-intentioned verbal explanations and may express their anxiety through nonverbal communication. Herein we discuss a case in which favorable results were obtained when surgical treatment was performed on a maxillary gingival carcinoma patient with mental retardation. The patient was a 61-year-old man who was admitted to our hospital with maxillary gingival pain. A biopsy revealed well-differentiated oral squamous cell carcinoma(cT4aN0M0, Stage ⅣA). Hemi-maxillectomy was performed and postoperative recovery was uneventful. A maxillary prosthesis was provided for oral rehabilitation. At the 5-year follow-up, there were no signs of recurrence or metastasis. This case illustrates the importance of collaboration between oral maxillofacial surgeons and specialists from related departments for the comprehensive management of patients with mental retardation.

Pindborg tumor associated with a supernumerary tooth: a case report

The calcifying epithelial odontogenic tumor is a rare benign neoplasm that accounts for approximately 1% of all odontogenic tumors. Most of the cases occur in the posterior mandible, and a few involve the maxilla. Despite their relatively indolent biological behavior, tumors in the maxilla tend to grow fast. We report the case of a 33-year-old female patient exhibiting swelling in the right maxilla. An isodense area associated with an impacted supernumerary tooth was found on imaging examination. The histopathologic diagnosis was a calcifying epithelial odontogenic tumor. The treatment of choice was surgical removal of the lesion and associated dental elements. The patient has been followed up for 11 months and shows no signs of recurrence. Besides describing this case, we reviewed the literature on the association of calcifying epithelial odontogenic tumors with supernumerary teeth and found two case reports addressing this subject.

Systematic review and meta-analysis of cervical metastases in oral maxillary squamous cell carcinoma.

BACKGROUND: Management of the node-negative neck in oral maxillary squamous cell carcinoma (SCC), encompassing the hard palate and upper alveolar subsites of the oral cavity, is controversial, with no clear international consensus or recommendation regarding elective neck dissection in the absence of cervical metastases. AIM: To assess the occult metastatic rate in patients with clinically node negative oral maxillary SCC; both as an overall metastatic rate, and a comparison of patients managed with an elective neck dissection at index surgery, compared to excision of the primary with clinical observation of the neck. METHODS AND RESULTS: A systematic review was performed by two independent investigators for studies relating to oral maxillary SCC and analysed according to PRISMA criteria. Data were extracted from Pubmed, Ovid MEDLINE, EMBASE, and SCOPUS via relevant MeSH terms. Grey literature was searched through Google Scholar and OpenGrey. Five hundred and fifty-three articles were identified on the initial search, 483 unique articles underwent screening against eligibility criteria, and 29 studies were identified for final data extraction. Incidence of occult metastases in patients with clinically node negative oral maxillary SCC was identified either on primary elective neck dissection or on routine follow up. Meta-analyses were performed. Of 553 relevant articles identified on initial search, 29 were included for analysis. The pooled overall rate of occult metastases in patients initially presenting with clinically node-negative disease was 22.2%. There is a statistically significant effect of END on decreasing regional recurrence demonstrated in this study (RR 0.36, 95% CI 0.24, 0.59). CONCLUSION: The results of this systematic review and meta-analysis suggest elective neck dissection for patients presenting with hard palate or upper alveolar SCC, even in a clinically node negative neck.

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.

Long-term Effect of Individualized Titanium Mesh in Orbital Floor Reconstruction After Maxillectomy.

OBJECTIVE: The aim of this study was to determine the clinical outcomes and long-term stability of individualized titanium mesh combined with free flap for orbital floor reconstruction after maxillectomy and to identify the risk factors for titanium mesh exposure. MATERIAL AND METHODS: The data of 66 patients who underwent maxillectomy and orbital floor defect reconstruction by individualized titanium mesh in Peking University School and Hospital of Stomatology between 2011 and 2019 were retrospectively reviewed. Postoperative ophthalmic function and success of aesthetic restoration were assessed. Titanium mesh exposure was recorded and the risk factors were identified. RESULTS: Mean follow-up was for 24.8 months (range, 6-92 months). Ophthalmic function was successfully restored in 63/66 patients. Aesthetic restoration was not considered satisfactory by 10 patients. Titanium mesh exposure occurred in six patients (exposure rate, 9.1%). Preoperative radiotherapy was identified as an independent risk factor for mesh exposure (OR = 28.8, P = 0.006). Previous surgery, postoperative radiotherapy, pathological type of the primary lesion, the type of tissue flap applied, and the use of intraoperative navigation were not significant risk factors. Six patients with titanium mesh exposure underwent second surgery, but mesh exposure recurred in two patients due to insufficient soft tissue coverage. CONCLUSION: Individualized titanium mesh with free flap can effectively restore maxilla-orbital defects. Preoperative radiotherapy is an independent predictor of postoperative titanium mesh exposure. Adequate soft tissue coverage of the mesh may reduce the risk of mesh exposure. LEVEL OF EVIDENCE: 4 (case-control study) Laryngoscope, 131:2231-2237, 2021.

Fully digital pathology laboratory routine and remote reporting of oral and maxillofacial diagnosis during the COVID-19 pandemic: a validation study.

The role of digital pathology in remote reporting has seen an increase during the COVID-19 pandemic. Recently, recommendations had been made regarding the urgent need of reorganizing head and neck cancer diagnostic services to provide a safe work environment for the staff. A total of 162 glass slides from 109 patients over a period of 5 weeks were included in this validation and were assessed by all pathologists in both analyses (digital and conventional) to allow intraobserver comparison. The intraobserver agreement between the digital method (DM) and conventional method (CM) was considered almost perfect (κ ranged from 0.85 to 0.98, with 95% CI, ranging from 0.81 to 1). The most significant and frequent disagreements within trainees encompassed epithelial dysplasia grading and differentiation among severe dysplasia (carcinoma in situ) and oral squamous cell carcinoma. The most frequent pitfall from DM was lag in screen mirroring. The lack of details of inflammatory cells and the need for a higher magnification to assess dysplasia were pointed in one case each. The COVID-19 crisis has accelerated and consolidated the use of online meeting tools, which would be a valuable resource even in the post-pandemic scenario. Adaptation in laboratory workflow, the advent of digital pathology and remote reporting can mitigate the impact of similar future disruptions to the oral and maxillofacial pathology laboratory workflow avoiding delays in diagnosis and report, to facilitate timely management of head and neck cancer patients. Graphical abstract.