Recurrent cementoblastoma with multifocal growth and cellular atypia: a case report.

BACKGROUND: Cementoblastoma is a rare odontogenic tumor characterized by the formation of osteocementum-like tissue on a tooth root directly by neoplastic cementoblasts. Although it is categorized as benign, it has a high potential for growth with a certain degree of recurrence risk. However, there are only a few studies describing the features of recurrent cementoblastoma. The diagnosis of recurrent cementoblastoma is challenging not only due to its cytological atypia but also because of its large size and multicentric growth pattern. These characteristics suggest a potential for malignancy. CASE PRESENTATION: A 29-year-old woman was transferred to our university dental hospital complaining of swelling of the right mandible. She had a history of enucleation of cementoblastoma associated with the third molar of the right mandible. Five years after the initial treatment, imaging demonstrated well-circumscribed multicentric radiopaque lesions in the same area. Histologically, the lesion consisted of osteocementum-like tissue rimmed with polygonal or plump tumor cells. Several cells were large epithelioid cells with bizarre nucleoli, which may be reminiscent of malignant tumors. Otherwise, there were no apparent malignant findings, including proliferative activity or atypical mitotic figure. Besides, tumor cells were positive for c-FOS, a marker of osteoblastoma and cementoblastoma. Eventually, the patient was diagnosed with recurrent cementoblastoma. CONCLUSIONS: Pathological analyses of this case suggested that the recurrent event in the cementoblastoma altered its growth pattern and tumor cell shape. Moreover, in the case of enucleation surgery, long-term follow-up is important because there is some recurrent risk of cementoblastoma, although it is not high.

Curettage combined with decompression for the treatment of ameloblastoma in children: report of two cases.

BACKGROUND: Ameloblastoma (AM) is the most common benign odontogenic tumor, which is more often detected in the mandible than maxilla, especially the mandibular body and mandibular angle. Pediatric AM is a rare disease, especially in patients aged 10 and younger. Compared with the mainstream osteotomy and reconstructive surgery for adult ameloblastoma, there is more room for discussion in the treatment of pediatric ameloblastoma. The postoperative functional and psychological influence can not be ignored. Especially for children in the period of growth and development, an osteotomy is often challenging to be accepted by their parents. We report two patients with ameloblastoma under 10 years old who are treated with curettage and fenestration, which is a beneficial method for children with ameloblastoma. CASE PRESENTATION: We present two cases of classic ameloblastoma in children. We describe in detail the patients' characteristics, treatment processes, and follow-up result. The bone formation and reconstruction in the lesion area after fenestration decompression and curettage are recorded at every clinic review. The surgical details and principles of curettage and decompression are also described and discussed. The two patients have good bone shape recovery and no recurrence. CONCLUSIONS: Children are in the growth and development period and possess an extremely strong ability of bone formation and reconstruction. Based on the principles of minimally invasive and functional preservation, we believe that curettage combined with decompression can be the first choice for treating AM in children, especially for mandibular lesions.

Long-Term Outcomes after Pediatric Mandibular Reconstruction Using Vascularized Free Fibula Flap.

BACKGROUND: The purpose of this study was to evaluate the long-term outcomes of mandibular reconstruction with vascularized free fibula flap in pediatric patients. METHODS: Consecutive cases of mandibular reconstruction with vascularized free fibula flaps in pediatric patients at Peking University School and Hospital of Stomatology between 1999 and 2019 were reviewed. Postoperative computed tomography (CT) data of all patients were collected at each postoperative follow-up point, and after the age of 18 years. The length and height of the grafted fibula and the length of the remaining mandible were evaluated by measuring the three-dimensional CT data using ProPlan CMF 3.0 software. Lower limb function was evaluated using the Enneking evaluation scale. Facial symmetry was self-evaluated and scored. Statistical analysis was performed on the data obtained. RESULTS: Fourteen patients were included in this study. All flaps were successful. The CT measurement results showed growth in the length of the grafted fibula that reconstructed the mandibular ramus and the residual mandible ( P < 0.05). The height of the grafted fibula remained stable ( P > 0.05). Eight patients were followed up until they were older than 18 years, and the CT measurement results after 18 years showed an essentially symmetric mandible profile ( P > 0.05). All patients were satisfied with their postoperative facial symmetry. Enneking evaluation scores showed good recovery of lower limb functions. CONCLUSIONS: The vascularized free fibula flap for mandibular reconstruction in pediatric patients is safe and reliable. It also provides good cosmetic and functional outcomes, as it demonstrated positive growth. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

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.

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.

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.

Radical resection of mandibular ameloblastoma and functional reconstruction with a fibula free flap. Report of two cases and review of the literature.

INTRODUCTION: Ameloblastoma is a borderline bone tumor that origins from the residual epithelium of the teeth germs, the epithelium of the enamel organ or the epithelium of odontogenic cysts. Ameloblastoma management is challenging owing to the necessity of tumor radical excision and the functional and aesthetic reconstruction of the surgical defect. The fibula-free flap (FFF) provides a high-quality and predictable mandibular reconstruction due to the high-caliber vascular pedicle, the bone length that can reconstruct large defects, the possibility for implants-based prosthetic reconstruction, and the possibility of harvesting a composite flap that can replace the mucosa, hence protecting the underlying bone reconstruction. CASE REPORTS: We report adult female and elder male patients, who were addressed to our hospital for mandible swelling and histopathological results of ameloblastoma. The lesions were treated by segmental mandibulectomy and FFF reconstructions. Osteosynthesis plates and screws were enough for the female patient's reconstruction of the lateral mandible defect and a load-bearing plate was necessary for the male patient's reconstruction of the surgical defect that included the anterior part of the mandible. The facial artery was used in both cases, and the surgeries lasted approximately 8 hours. No recurrence was observed at the follow-up and the aesthetic function was well re-established. CONCLUSION: Radical treatment of ameloblastoma is mandatory. The aesthetic function could be properly maintained by FFF. Also, the FFF reconstruction is a reliable method for head and neck large bone and soft tissue defects, microvascular anastomosis on facial artery offering a good blood SUPPLY OF THE FLAP. KEY WORDS: Ameloblastoma, Fibula-free flap, Maxillofacial reconstruction, Radical treatment.

Giant Mandibular Ameloblastoma with Rare Hypercalcemia: A Case Report and Literature Review.

Ameloblastoma is the most common benign odontogenic tumor with local invasion and high recurrence, which generally occurs in the jaw bones. Hypercalcemia is a common paraneoplastic syndrome that is commonly observed in patients with malignancies but rarely encountered in patients with benign tumors. Thus far, not many cases of ameloblastoma with hypercalcemia have been reported, and the pathogenic mechanism has not been studied in depth. This paper presents a case report of a 26-year-old male diagnosed with giant ameloblastoma of the mandible, accompanied by rare hypercalcemia. Additionally, a review of the relevant literature is conducted. This patient initially underwent marsupialization, yet this treatment was not effective, which indicated that the selection of the appropriate operation is of prime importance for improving the prognosis of patients with ameloblastoma. The tumor not only failed to shrink but gradually increased in size, accompanied by multiple complications including hypercalcemia, renal dysfunction, anemia, and cachexia. Due to the contradiction between the necessity of tumor resection and the patient's poor systemic condition, we implemented a multi-disciplinary team (MDT) meeting to better evaluate this patient's condition and design an individualized treatment strategy. The patient subsequently received a variety of interventions to improve the general conditions until he could tolerate surgery, and finally underwent the successful resection of giant ameloblastoma and reconstruction with vascularized fibular flap. No tumor recurrence or distance metastasis was observed during 5 years of follow-up. Additionally, the absence of hypercalcemia recurrence was also noted.

Curettage combined with bone cavity opening reduces recurrence of the mandibular conventional ameloblastoma and effectively preserves the mandible: a retrospective study.

BACKGROUND: Patients with mandibular conventional ameloblastoma undergoing radical surgical treatment experience greater trauma and often find it challenging to accept, whereas conservative therapy is associated with a higher recurrence rate. In this study, we have improved traditional conservative treatment for mandibular conventional ameloblastoma by curettage combined with bone cavity opening (Cur/BCO). This retrospective study aimed to evaluate the effectiveness of the Cur/BCO treatment by comparing its recurrence rate and bone mineral density (BMD) growth rate with the traditional conservative treatment approach. METHODS: A total of 40 patients, meeting the study's inclusion and exclusion criteria from 2012 to 2020, were screened, with 20 in the modified group and 20 in the traditional group. ImageJ (RRID: SCR_003070) software was employed for measuring image indices. All data were analyzed using T-test, Chi-square test and Fisher exact test in SPSS 26.0 (p = 0.05). RESULTS: The incidence of recurrence was significantly lower in the modified group, at only 5%, compared to 35% in the traditional group (p < 0.05). Regarding bone mineral density (BMD) growth rate, the average value in the modified group was 0.0862 ± 0.2302 (/month), significantly higher than the average value of 0.0608 ± 0.2474 (/month) in the traditional group (p < 0.05). CONCLUSIONS: In this study, it was found that the recurrence rate of the modified conservative treatment (Cur/BCO) was lower than that of the traditional conservative treatment for managing mandibular conventional ameloblastoma. Furthermore, the BMD growth rate was quicker in the modified group. Thus, Cur/BCO could be considered as a viable option for the conservative treatment of mandibular conventional ameloblastoma.

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.

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.

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.

Planning of physiotherapeutic procedure in patients after mandible reconstruction taking into account donor site: a literature review.

BACKGROUND: Mandible tumors are very rare. One of the main methods of the treatments is resection of the tumor and then reconstruction of the mandible. The donor site is often distant tissue-fibula or ilium. Following this, it is necessary to improve the patient in two ways, on one hand restoring the function of the mandible, and on the other hand, improving the donor site area. For that reason, physiotherapy after tumor resection and reconstruction of the mandible is very complicated. The aim of this bibliographic review was to find the methods of the reconstruction of the mandible in the context of patients' functional assessment after surgeries to create effective physiotherapeutic procedures in the feature. METHODS: PEDro, Medline (PubMed), Cochrane Clinical Trials were searched. RESULTS: 767 articles were found. 40 articles were included to this literature review. CONCLUSIONS: Authors showed different kinds of surgeries strategy for patients with tumors of the mandible. They also showed manners of patients' functional assessment in the localization of transplantation and donor site. It could be useful for physiotherapists during planning of comprehensive physiotherapy.

Symmetrical Midfacial Growth After Pediatric Mandibular Reconstruction With Free Fibula Flap.

BACKGROUND: Free fibula is the workhorse flap for mandibular reconstruction and is increasingly being used in pediatric patients. However, craniomaxillofacial growth and development involve interdependent processes, and it remains unknown whether mandibular reconstruction with free fibula allows symmetric growth of the midface. PURPOSE: The study evaluated midfacial symmetry after pediatric mandibular defect reconstruction. STUDY DESIGN, SETTING, SAMPLE: This retrospective cohort study included pediatric patients aged ≤14 years who underwent mandibular reconstruction with free fibula flap. Postoperative computed tomography data were obtained at predefined follow-up time points. Midfacial symmetry was evaluated based on 3-dimensional (3D) cephalometry. PREDICTOR VARIABLE: The predictor variable was the side of the midface (affected or healthy side relative to the mandibular defect). MAIN OUTCOME VARIABLES: The primary outcome variable was postoperative midfacial symmetry (at 1 week, 6 months, 1 year, 2 years, and >3 years, or after the age of 18 years), assessed in horizontal, vertical, and anteroposterior dimensions using 3D cephalometry. Another outcome variable was patient satisfaction based on a self-evaluation using visual analog scoring. COVARIATES: Sex, age, diagnosis, and type of denture restoration. ANALYSES: Paired t tests were performed to assess the relationship between the predictor and outcome variables, with the significance level of P < .05. RESULTS: A total of 13 patients were included in this study (9 males and 4 females; mean age: 12.23 ± 2.39 years). The average distance from upper first molar point (U6) to the horizontal plane on the affected side became greater than on the healthy side (difference: 0.7 ± 0.5 mm to 1.6 ± 1.4 mm, P < .05), while the average distance from pterygomaxillary fissure to coronal plane on affected side became shorter than that on the healthy side (difference: 0.6 ± 0.6 mm to 1.2 ± 1.1 mm, P < .05) from 1 year after the surgery. There were no statistically significant differences in the remaining measurements between the 2 sides (P > .05). All the patients were satisfied with their postoperative facial symmetry. CONCLUSIONS AND RELEVANCE: There were no severe midface deformities after pediatric mandibular reconstruction with free fibula flap. Meanwhile, pediatric mandibular reconstruction and proper occlusion could promote midfacial growth and symmetry.

Impact of fibula osteoseptocutaneous flap laterality in mandibular and intraoral reconstruction.

BACKGROUND: The fibula flap has been the workhorse flap for mandibular reconstruction. However, relationships among the bone, skin, and vessels raise concerns about donor-side selection. This study aimed to clarify its impact on clinical outcomes. METHODS: Between September 2013 and June 2021, 61 cases of fibula osteoseptocutaneous flaps for mandibular and intraoral reconstruction were categorized into the landing-down (N = 25) and swing-up (N = 36) groups depending on whether the skin was easily accessible within the oral cavity. The demographics, operative findings, and outcomes of the cases were compared. RESULTS: Overall, seven (11%) flaps developed skin necrosis, including four partial and three total necrosis. The skin necrosis rate was higher in the swing-up than in the landing-down group (19% vs. 0%, p = 0.035). CONCLUSIONS: When using the fibula osteoseptocutaneous flap for mandibular and intraoral reconstructions, ensuring that the skin is properly located within the oral cavity could reduce the risk of skin necrosis.

A novel alloplastic grid reconstruction plate for the mandible - Retrospective comparative clinical analysis of failure rates and specific complications.

PURPOSE: This study aimed to investigate the effect of three different osteosynthesis plate systems on failure rates and complications after continuity-interrupting mandibular resections with alloplastic reconstructions. MATERIALS AND METHODS: Records of respective patients from 2010 to 2020 were analyzed retrospectively. The analyses included the osteosynthesis plate type (2.4 MANDIBULAR (RP1: MANDIBULAR [Medicon®, Tuttlingen, Germany]; RP2: Modus® Reco 2.5 [Medartis®, Basel, Switzerland]; and RP3: Modus 2 Mandible [Medartis®, Basel, Switzerland]), extent & location of the defect, age, sex, radiotherapy, and nicotine abuse. In case of failure, timepoint, and the problem, namely oral/extraoral dehiscence, screw loosening, and plate fractures that led to removal, were analyzed. Complications were classified according to Clavien-Dindo system. RESULTS: A total of 136 patients were included. The mean follow-up time was 18 ± 26 months. Survival rates after 1, 2, and 5 years were 69.9%, 66.9%, and 64.7%, respectively. Although survival was not significantly associated with the reconstruction system, the most frequent complications were seen in cases of RP1 & RP2 when compared to RP3 (p = 0.033). In brief, dehiscences were seen significantly less often in cases of RP3 (12.5%) when compared to RP1 (44.7%) and RP2 (26.9%; p = 0.024). Fractures of the osteosynthesis systems occurred in 3 of 4 cases (75%) with RP1, in 1 of 4 cases (25%) using RP2, and in no single case using the RP3 system (p = 0.03). Most of the observed complications occurred up to 12 months postoperatively. A total plate survival rate of 64.7% and a total plate complication rate of 47.8% were seen. CONCLUSION: In conclusion, it seems that RP3 should be preferred over RP1 and RP2 regarding failure rates and complications.