Ameloblastic fibrosarcoma of the maxilla arising in an old woman, a rare case report and literature review.

BACKGROUND: Ameloblastic fibrosarcoma (AFS) is a rare malignant odontogenic tumor, commonly occurring in young adults and typically affecting the mandibular region. We report an exceptionally rare and highly atypical case of AFS in an elderly female patient originating from the maxillary bone. CASE PRESENTATION: A 66-year-old woman was admitted with a two-week history of a lump in her left upper molar. CT scans suggested a cyst in the maxillary bone. An incisional biopsy revealed a spindle cell neoplasm. MRI showed abnormalities in the left maxilla, indicating a possible tumorous lesion. The patient underwent a subtotal maxillectomy, wide tumor excision, intraoral epithelial flap transplantation, and dental extraction. Histology identified atypical tumor cells with visible mitotic figures. Immunohistochemistry showed negative for PCK and CD34 expression, but positive for Vimentin and SMA expression. The Ki-67 proliferation index ranged from 30 to 50%. These findings suggested a potentially malignant soft tissue tumor in the left maxilla, leaning towards a diagnosis of AFS. The patient received postoperative radiotherapy. There was no recurrence during the six-month follow-up. CONCLUSION: Based on repeated pathological evidence, we report a rare case of an elderly female with AFS originating from the maxillary bone. Surgery and postoperative radiotherapy resulted in a favorable outcome.

Central Myoepithelioma of the Maxilla.

Myoepithelioma is a benign salivary gland tumor. Central myoepitheliomas are very rare. The aim of this report was to describe a case of maxillary myoepithelioma. A 14-year-old female patient presented with an multilocular lesion in the anterior maxilla, with nearly 8 months of duration. The lesion was asymptomatic, and the patient's dental history was unremarkable. The diagnostic hypothesis was an odontogenic tumor. Biopsy specimen consisted of nests of plasmacytoid cells in a myxoid stroma without duct formation. No cellular atypia or bone and cartilage formation were noted. The neoplastic cells were positive for Pan-cytokeratin, S100, CK7, and CK8. The final diagnosis was myoepithelioma. The patient was treated by surgical excision followed by bone curettage, and no signs of recurrence were found after 8 years of treatment.

Prosthetic rehabilitation of patients with maxillary oncology defects using zygomatic implants.

PURPOSE: Prosthetics for patients after oncological resection of the upper jaw is a complex problem associated with the physiological and anatomical separation of the oral cavity and the nasal/paranasal region. This study reports the clinical results of the use of the zygomatic implants for prosthetic rehabilitation in patients with maxillectomy due to upper jaw tumors. MATERIALS AND METHODS: The study included 16 patients who underwent prosthetic rehabilitation using a zygomatic implant after maxillectomy period from 2021 to 2023. After the tumor was removed, immediate surgical obturators were placed. Main prosthetic rehabilitation was performed 6-12 months after tumor removal, but before that, a temporary obturator was made and used. Six-twelve months after tumor resection, 1-4 zygomatic implants were inserted into the zygomatic bone unilaterally or bilaterally. A total of 42 zygomatic implants were installed, 2 of which were unsuccessful and were removed in 1 patient. The implants were placed using the surgical guide, which was planned and prepared digitally. RESULTS: No postsurgical complications were seen, and the patients were discharged from the hospital after 7-10 days. The patients were able to return to a normal diet (hard food) after just 7 days following surgery, with no further complaints regarding function or pain, apart from the residual edema caused by the intervention. CONCLUSIONS: The use of prostheses fixed on zygomatic implants in patients with maxillary defects is an effective method of prosthodontic rehabilitation in complex clinical cases after maxillectomy.

[Komplexes Odontom im Unterkieferfrontzahnbereich bei einer 16-jährigen Patientin - Diagnostik, Therapie und Nachsorge].

Odontome gelten zusammen mit den Amelo- blastomen als die häufigsten odontogenen Tumoren. Sie entstehen während der embryo- nalen Zahnkeimentwicklung durch fehlerhaft differenziertes Keimgewebe und werden daher auch als Hamartome bezeichnet. Somit sind sie also strenggenommen keine klassischen Neoplasien.

Solitary intraosseous neurofibroma of the oral cavity: rare localization in the maxilla.

BACKGROUND: Neurofibroma is a common benign tumor of neuronal origin that can occur as a solitary tumor or as a component of the generalized syndrome of neurofibromatosis. Neurofibromas are primarily located in the subcutaneous soft tissues and commonly involve extra-oral sites. Solitary intraosseous neurofibromas of the oral cavity are infrequent, with occurrences in the maxilla being exceedingly rare. CASE PRESENTATION: A 22-year-old male patient presented with an asymptomatic mass in the maxilla. Cone-beam computed tomography revealed a round, well-outlined, radiolucent lesion with expansive growth. The neoplasm with the complete capsule was completely removed and confirmed as a neurofibroma based on histopathological and immunohistochemical findings. The reported cases of solitary intraosseous neurofibromas located in the maxilla published in the English literature were compiled to assist in the diagnosis of solitary intraosseous neurofibromas of the maxilla. Nine months after the surgery, there were no signs of tumor recurrence or malignant transformation. CONCLUSIONS: This report emphasizes that rare locations of neurofibromas, such as solitary intraosseous neurofibromas in the maxilla, typically demonstrate nonspecific clinical and radiological features. Clinicians should consider solitary intraosseous neurofibromas as possible differential diagnoses and recognize the histopathological and immunohistochemical features to confirm the correct diagnosis. A longer follow-up period is required because of the potential for local recurrence and malignant transformation of these tumors.

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.

Surgical Resection and Reconstruction of Ameloblastoma: A 13-Year Retrospective Review.

BACKGROUND: Ameloblastoma is a locally aggressive, benign tumor presenting in the maxilla and mandible prone to recurrence. Resection greatly limits recurrence; however, reconstruction becomes critical to preserve patients' functionality and esthetics. PURPOSE: The aim of this study was to describe surgical resection and reconstructive approaches in the treatment of ameloblastoma and compare clinical outcomes to conservative methods of treatment. STUDY DESIGN, SETTING, SAMPLE: A retrospective case series was completed through analysis of patient records. The study population was composed of patients treated for ameloblastoma at the Royal Brisbane Hospital (Queensland, Australia) in the Oral and Maxillofacial Surgery Unit from January 1, 2008, to December 31, 2020. Patients without histological confirmation of intraosseous ameloblastoma were excluded from the study sample. PREDICTOR VARIABLE: Not applicable. MAIN OUTCOME VARIABLE(S): The primary outcome variable was time to recurrence. Secondary outcome variables included any surgical complications incurred. COVARIATES: The covariate variables collected included age at diagnosis/treatment, gender, ethnicity, location of lesion and site(s) of involvement, tumor extent, alveolar expansion, histopathological growth pattern, and soft tissue involvement. ANALYSES: Descriptive statistics were computed for each study variable. RESULTS: A total of 48 cases of histologically confirmed ameloblastoma were identified (41 mandibular, 7 maxillary) involving 50 excisional operations (44 resections, 6 enucleations). Of these cases, 44 were followed up > 12 months, with a mean length of follow-up time of 65.6 months. No recurrence was detected for resected lesions. One enucleated lesion recurred at 25 months. Thirty-seven reconstructive procedures were undertaken, including 32 immediate free flaps. All reconstructive flaps and grafts survived, and no major complications were recorded. CONCLUSION AND RELEVANCE: Resection of ameloblastoma limits recurrence and should be considered curative. Immediate microvascular free flap reconstruction of maxillary and mandibular defects from resection of ameloblastoma is safe and predictable.

Quality of life in oncologic patients after maxillectomy operations: clinical case series on different rehabilitation protocols.

OBJECTIVE: The study's purpose was to compare the quality of life (QoL) in oncologic patients treated with different rehabilitation protocols following maxillary tumor resections. PATIENTS AND METHODS: The patients were divided into three groups. Group A: 18 Patients with maxillary obturator prosthesis. Group B: 17 Patients with simultaneous autologous tissue reconstruction. Group C: 12 Patients with prosthesis on zygomatic implants. The post-operative QoL was compared using standard questionnaires, investigating items like pain, mood, social relations, and specific functions that could potentially compromise the post-operative QoL. A secondary analysis compared reconstructed vs. non-reconstructed patients. RESULTS: Most questionnaire items did not show significant differences among groups. Statistically significant outcomes were found in two parameters (social contact and sexuality), in which patients treated with zygomatic implants had the best satisfaction, and patients with obturator prostheses showed the lowest satisfaction. Patients belonging to the non-reconstructed group showed better moods than those in the reconstructed group, while taste problem complaints and pain were lower in the reconstructed group. CONCLUSIONS: Although the type of reconstruction procedure depends on the type of maxillectomy to be performed and on the general health situation of each patient, the impact of the rehabilitation protocol on the patients' QoL should be accounted for when planning the treatment.

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.

Virtual Surgical Guidance Improves Quality of Life Following Scapular Free-Flap Reconstruction of Maxillary Defects.

BACKGROUND: The best approach to maxillary reconstruction with negative impact on the patient's quality of life (QOL) remains the subject of debate. PURPOSE: This study was designed to evaluate the QOL outcomes following maxillary reconstruction using a scapular-free flap, with and without the computer-aided design and computer-aided manufacturing (CAD/CAM) technique. STUDY DESIGN, SETTING, SAMPLE: A prospective randomized controlled clinical trial was performed following the CONSORT checklist. To be included, patients suffering from maxillary defects without previous surgery or lymph node involvement were selected. Patients with poor oncological prognosis, deficient performance status, preoperative chemotherapy, or radiotherapy, besides vascular contraindications, were excluded. PREDICTOR VARIABLE: The predictor variable was the reconstruction technique, grouped into two equal categories. Patients were randomly assigned to reconstruction with a scapular-free flap, with the control group not utilizing a guide and the study group utilizing a CAD/CAM-customized guide. MAIN OUTCOME VARIABLE: The primary outcome variable was the various domains of QOL, measured using the University of Washington Quality of Life score, 12 months after reconstruction. COVARIATES: Age; sex; scapular flap side; and maxillary defect tumour type, histopathology, side, length, class, sagittal, and axial differential areas were considered. ANALYSES: Shapiro-Wilk and Kolmogorov-Smirnov tests were performed for normality. For comparisons between the two groups, Student's t-test and Mann-Whitney test were used. The category variables were compared using the χ2 test. P ≤ .05 was considered statistically significant. RESULTS: The sample was composed of twenty-two eligible patients, eleven in each group. The mean age of the control group was (50.09 ± 17.14) years, and 45.45% were male, while in the study group, the mean age was (48.36 ± 14.14) years, with 36.36% male. All the patients were evaluated, showing statistically significant differences between control and study groups in terms of pain, appearance, chewing, swallowing, speech, and shoulder (P ≤ .05), indicating that the scapular-free flap with a CAD/CAM osteotomy guide had improved the patient's QOL. CONCLUSION AND RELEVANCE: The findings of this study indicate that virtual surgical planning with a CAD/CAM customized osteotomy guide using a scapular-free flap can improve QOL in maxillary reconstruction in terms of pain, appearance, chewing and swallowing food, pronouncing words, and shoulder QOL outcomes.

Reconstruction of Extensive Maxillary Defects Using Flow-Through Fibula Free Flap With Anterolateral Thigh Free Flap.

BACKGROUND: The maxillary defects left unreconstructed or inadequately reconstructed often result in significant functional and esthetic impairments. Adequate reconstruction of extensive maxillary defects requires a sufficient volume of hard and soft tissues. METHODS: A 48-year-old male presenting bilateral extensive maxillary defects underwent secondary reconstruction with a flow-through fibula free flap in combination with an anterolateral thigh free flap. RESULTS: The use of flow-through technique allowed minimizing the problem of limited recipient vessels and the length of free flap vascular pedicle usually encountered in secondary reconstruction. The bilateral maxillary defects were successfully reconstructed, and the postoperative outcomes were uneventful. The patient was satisfied with the treatment outcomes. He is being followed up and was referred to the implantology department for the placement of osseointegrated dental implants. CONCLUSIONS: The flow-through fibula free flap, in combination with the anterolateral thigh free flap, was found reliable and feasible for this case of secondary reconstruction of bilateral maxillary defects. This technique has provided satisfactory functional and esthetic outcomes and effectively improved the patient's self-esteem.

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.

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.

Transoral versus transfacial surgical approach to maxillary tumors: evaluation of outcomes and perspectives.

Neoplasms of the maxilla have multiple different origins and histology, and often extend towards the infratemporal fossa, orbit, or skull base. Extensive resection may be required, often leading to poor esthetic and functional results. Usually, these lesions are removed via a transfacial approach. The aim of this study was to compare the outcomes of the transoral versus transfacial approach for maxillary tumors. A single-institution retrospective study was conducted on patients with maxillary-midface tumors, treated between January 2009 and December 2019. The patients were divided into two groups according to the surgical approach, transfacial or transoral, and the following outcomes were assessed: extent of the resection based on Brown's classification; postoperative pathology margin assessment; reconstruction technique; esthetic/functional results. A total of 178 patients were included. A satisfactory resection was obtained in both groups, with the transoral cohort achieving a higher rate of clear oncological margins (positive margins: transoral group 3.7% versus transfacial group 6.8%, P = 0.389) and a significantly higher University of Washington Quality of Life score (mean 72.2 versus 67.8, P < 0.001). Even large and invasive tumors can be treated successfully with the transoral approach, avoiding unesthetic facial scars while still providing complete resection of the tumor.

Ghana 3D Telemedicine International MDT: A proof-of-concept study.

A real-time 3D Telemedicine system - leveraging Microsoft's Holoportation™ communication technology - enabled an international multidisciplinary team meeting (MDT) to consult with complex reconstructive patients before, during, and after an overseas surgical collaboration. METHODS: A proof-of-concept international 3D MDT clinic took place in November 2022, between the Canniesburn Plastic Surgery Unit, UK, and the National Reconstructive Plastic Surgery and Burns Centre, Korle Bu Teaching Hospital, Ghana. The 3D system was utilised 1) previsit to assess patients and enable logistical planning, 2) on-site in Ghana to further allow patients to see themselves and proposed operations in 3D, and 3) post visit to debrief the team and patients. RESULTS: Four Ghana patients were followed through their patient journey (mandibular ameloblastoma, sarcoma thigh, maxillary tumour, sarcoma back). Thirteen participants (four patients, four Ghana clinicians, and five UK clinicians) completed feedback on the 3D MDT. Outcome measures were rated highly with satisfaction 84.31/100, perceived benefit 4.54/5, overall quality 127.3/147 (Telehealth Usability Questionnaire), and usability 83.2/100 (System Usability Scale). These data show close alignment with that previously published on high-income countries. CONCLUSIONS: This novel technology has the potential to enhance the delivery of overseas surgical visits to low-to-middle-income countries, by improving planning, informed discussion with patients, expert consensus on complex cases, and fostering engagement with professionals who may be thousands of miles away. This is the first demonstration that real-time 3D Telemedicine can both work, and enhance care within an international MDT clinic, and may thus enable change in the approach to overseas surgical collaborations.

Surgical and health related quality of life outcomes following treatment with zygomatic implant perforated (ZIP) flaps.

BACKGROUND: The zygomatic implant perforated (ZIP) flap is a novel approach to the challenge of reconstructing the maxilla. We report on our experience using the ZIP flap technique for patients undergoing infrastructure maxillectomy at Chris O'Brien Lifehouse, Sydney, Australia. METHODS: Thirteen patients who underwent a ZIP flap reconstruction between August 2019 and August 2021 were identified. Demographic, surgical, and histopathological information was collected. Health Related Quality of Life (HRQOL) was assessed using the FACE-Q Head and Neck Cancer module, the M.D. Anderson Dysphagia Inventory, and the Speech Handicap Index. RESULTS: A total of 44 zygomatic implants were placed, of which 42 (95%) survived. The median time from surgery to dental rehabilitation was 35 days. HRQOL data was available for nine patients over 24 months, demonstrating improved speech and swallowing outcomes over the follow up period. CONCLUSIONS: The ZIP flap is a reproducible surgical technique that facilitates rapid dental rehabilitation post infrastructure maxillectomy.