Carbon-ion radiotherapy for clear cell odontogenic carcinomas.

BACKGROUND: Clear cell odontogenic carcinoma (CCOC) is a rare odontogenic malignant tumor. The standard treatment for CCOC is surgical resection and adjuvant radiotherapy (RT). Radiotherapy is generally considered in inoperable cases. However, there are no reports on definitive RT for CCOC, and the role of RT in patients with inoperable CCOC remains unknown. Therefore, in this report, we present two cases of carbon-ion (C-ion) RT for CCOC. CASE PRESENTATION: In case 1, a 73-year-old man with mandibular CCOC presented with recurrence in the inferior temporal fossa after two tumor resections. The tumor was considered inoperable, and C-ion RT (57.6 Gy in 16 fractions) was administered. The tumor remained controlled even after 20 months of C-ion RT; however, the patient died of other causes. In case 2, a 34-year-old man with maxillary CCOC presented with recurrence in the left sinonasal region after two tumor resections. The tumor was considered inoperable, and C-ion RT (64 Gy in 16 fractions) was administered. However, recurrence was observed in the irradiated field 19 months after the treatment. Subsequently, C-ion RT (64 Gy in 16 fractions) was repeated for the recurrent tumors. Seven years and 6 months after the initial irradiation, the tumor remains controlled, and the patient is alive without any unexpected serious adverse events. CONCLUSION: C-ion RT may be an effective treatment option for patients with inoperable CCOC.

Intraosseous myofibroma mimicking an odontogenic lesion: case report, literature review, and differential diagnosis.

BACKGROUND: Intraosseous myofibroma of the jaw is a rare neoplasm of mesenchymal origin with limited comprehensive understanding. It typically affects patients in the first two decades of life with a male predilection. CASE PRESENTATION: This study presents a rare case of myofibroma mimicking an odontogenic lesion in a 2-year-old boy. The patient presented with an incidental finding of a painless swelling of the right mandibular ramus of unknown etiology. Imaging analysis revealed a solid, expansile lesion adjacent to the germinal zone of the right mandibular first molar. Histopathologic analysis and immunohistochemistry after incisional biopsy suggested a possible central odontogenic fibroma, and the patient underwent total enucleation, leading to the final diagnosis of intraosseous myofibroma. Follow-up examinations showed no evidence of recurrence. CONCLUSIONS: This report contributes to the understanding of myofibroma in pediatric patients and underscores the critical role of meticulous histopathologic examination for effective surgical planning and optimal patient outcomes.

Aggressive Late Recurrence of a Calcifying Epithelial Odontogenic Tumor: Surgical Refinements Based on New Technologies.

A calcifying epithelial odontogenic tumor is a rare, benign odontogenic neoplasm. Surgical treatment is the option, and late recurrence is very rare. Radiologically, the lesions are commonly present scattered calcifications. This case report details a 64-year-old female patient with a recurrence of a right mandibular calcifying epithelial odontogenic tumor 2 decades after successful initial surgical removal. A segmental mandibulectomy and immediate reconstruction were performed using a planned vascularized free fibula flap with virtual surgery, custom reconstruction plate, and intraoperative computed tomography. Modifications were made to the design of the reconstruction plate to improve the cervicofacial profile and subsequent rehabilitation with dental implants. Fully guided implant surgery with point-of-care manufacturing protocol was done to improve prosthetically driven implant planning. The case presented highlights the usefulness of new technologies for mandibular reconstruction with the free fibula flap and the concept of point-of-care with technical notes that increase precision and reduce morbidity in implant-supported rehabilitation.

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

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

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.

What Is the 5-year Incidence of Recurrent Disease of Odontogenic Keratocysts?

PURPOSE: There is no consensus on the ideal treatment of odontogenic keratocysts (OKCs). Certain risk factors may modify the disease recurrence rate. The purpose of this study was: 1) to estimate the incidence of OKC recurrence in a statistically rigorous manner; and 2) to identify risk factors associated with OKC recurrence. METHODS: The investigators designed and implemented a retrospective cohort study and enrolled a sample derived from the population of patients presenting to the Department of Oral and Maxillofacial Surgery outpatient clinics at the University of Washington School of Dentistry and Harborview Medical Center for evaluation and management of OKCs between January 1, 2010, and December 31, 2020. Predictor variables included demographics (age, gender), radiographic characteristics (location of lesion, locularity, size, and cortical perforation), and type of operation performed (decompression + cystectomy, enucleation ± adjuvant therapy, or resection). The primary outcome variable was time to recurrent disease, defined as the time from treatment to radiographic or clinical evidence of a new lesion (recorded in months). Kaplan-Meier analysis was used to estimate median time to recurrence, and Cox proportional hazards models were used to identify covariates statistically associated with recurrent disease (P ≤ .05). RESULTS: The sample was composed of 60 subjects with 63 previously untreated lesions. Eight subjects (13%) developed a recurrent lesion during the study interval with a median time to recurrence of 31 (interquartile range, 24 to 48) months. One of the 8 recurrences occurred within 12 months of treatment and 7 of the 8 recurrences occurred more than 21 months after treatment. Based on using Kaplan-Meier analysis, the 5-year incidence of disease recurrence was estimated to be 34%. Lesions with cortical perforation were 8.3 times more likely to recur (95% confidence interval [1.7, 41.3]; P-value = .01), and multilocular lesions were 10.6 times more likely to recur (95% confidence interval [1.3, 86.9]; P-value = .03). The sample size was the limiting factor to performing regression analyses. CONCLUSIONS: Virtually every publication on OKCs to date reports frequencies of disease recurrence rather than applying appropriate survival analyses commonly used to estimate outcomes in cancer research. Failure to use the appropriate statistical analyses underestimates the risk of disease recurrence. Our study is no exception. The estimated frequency of disease recurrence during the study interval was 13% (8/60). When survival analyses are applied that account for varying months of follow-up, the incidence of disease recurrence is 34 per 100 per 5 years. We recommend the application of time-to-event analyses in the study of disease entities with the potential for recurrence.

Diode laser on excision of giant peripheral odontogenic myxoma: a rare case report and literature review.

It was to report a rare case of peripheral odontogenic myxoma removed with high-power diode laser and to do an extensive review of studies of odontogenic cysts and tumors treated with high-power laser (HPL). This is a rare case of a 63-year-old male patient with a peripheral odontogenic myxoma measuring approximately 10 cm in the attached gingiva region of tooth 16 removed with a high-power diode laser (808 nm, 3 W, in continuous mode, under constant suction, with 400-µm optical fiber). A literature review was also carried out looking for articles that involved the use of HPL in the treatment of odontogenic cysts and tumors, without restriction of year or language. In the present case, there was no need for suturing, no postoperative discomfort, and minimal bleeding during the procedure. In a 12-month follow-up period, there were no signs of recurrence. Only two cases of intra-osseous odontogenic myxomas treated with HPL and 10 cases involving other odontogenic cysts and tumors were found. All studies showing HPL to be effective in treating these lesions. Despite the different types of lasers used and different parameters, it is observed that lasers are effective in the treatment of odontogenic lesions.

A Rare Case of a Large Ameloblastoma and Cervical Lynphoepitelial Cyst Treated Simultaneously by an Intraoral Approach and Immediate Reconstruction.

Ameloblastoma is a benign locally aggressive odontogenic tumor of epithelial origin with unlimited growth capacity. Cervical lymphoepithelial cyst is a developmental cyst located in the superior-lateral region of the neck with treatment consisting of surgical excision, which presents low recurrence rates. This article reports the case of patient J.G.S.L., 24 years old, female, who reported to the service with a histopathological evaluation compatible with intraosseous mandibular ameloblastoma. On physical examination, a nontender swelling was palpable along the body of left mandible where the ameloblastoma was located, with a fluctuant region detected just below it. Imaging exams confirmed the suspicion of two independent lesions. The treatment plan consisted of two stages: first, mandibular resection of the area affected by the ameloblastoma, followed by reconstruction with a titanium plate simultaneous to excisional biopsy of the as yet undiagnosed lesion, both through intraoral access; second, mandibular reconstruction with iliac crest graft. Histopathological examination of the second lesion indicated a diagnosis of cervical lymphoepithelial cyst, being the first case in the literature with these concomitant lesions. Currently, the patient is in her third postoperative year with no signs of recurrence.

Prominent osteolysis in the maxilla: case report of an odontogenic fibroma mimicking a cyst.

BACKGROUND: Odontogenic fibroma (OF) is a rare benign odontogenic tumor of ectomesenchymal origin, mostly affecting the tooth-bearing portions of the jaws in middle-aged patients. Whilst small lesions tend to be clinically asymptomatic, varying unspecific clinical symptoms occur with an increase in size and may mimic odontogenic or other maxillofacial bone tumors, cysts, or fibro-osseous lesions of the jaws. CASE PRESENTATION: A 31-year-old female patient presented with a hard, non-fluctuating protrusion in the vestibule of the upper right maxilla. It was visualized on cone beam computed tomography (CBCT) as space-occupying osteolysis with the displacement of the floor and facial wall of the maxillary sinus, mimicking a cyst-like lesion. The tissue was surgically removed and identified as an OF in the histopathological examination. One year after the surgery, restitution of regular sinus anatomy and physiological intraoral findings were observed. CONCLUSIONS: This case report emphasizes that rare entities, like the maxillary OF presented, often demonstrate nonspecific clinical and radiological findings. Nevertheless, clinicians need to consider rare entities as possible differential diagnoses and plan the treatment accordingly. Histopathological examination is essential to conclude the diagnosis. OF rarely recur after proper enucleation.

A rare primordial odontogenic tumor in a pediatric patient.

Primordial odontogenic tumor (POT) is a rare, benign pediatric lesion that was first included in the 2017 edition of WHO Classification of Head and Neck Tumours. At that point, only 7 cases had been described in the literature; a total of 23 cases have been reported to date. This case report adds to that total by describing a case of POT in a 12-year-old girl and reviews the characteristics of this neoplasm in order to better characterize its clinical, radiographic, and histologic features and increase dentists' awareness of POT. The presence of calcifications within the lesion was reported in 4 of 23 previous cases, and calcifications were also observed in the current case. The presence of calcifications in POT is uncommon and may make radiographic diagnosis more challenging. Treatment of POT is simple excision, and recurrence after excision is rare. Accurate diagnosis is essential, as there has been at least 1 report of overtreatment involving mandibulectomy.

A Recurrent Case of Ameloblastic Fibroma in 37-year Old Male.

Ameloblastic fibroma (AF) is a benign mixed epithelial and mesenchymal odontogenic tumor. This was previously grouped in odontogenic tumor showing odontogenic epithelium with odontogenic ectomesenchyme, with or without hard tissue formation. This report describes a case of ameloblastic fibroma in a 37-yearold male who came with the complain of swelling in the left side of lower jaw since one year. Enucleation of the mass followed by reconstruction was done six years back. However, after two years of initial treatment; radiographic findings suggested recurrence. Histopathological examination confirmed the diagnosis of ameloblastic fibroma. Patient had no clinical and radiographic evidence of recurrence in three and six months' follow-up. Because of the higher proliferative capacity and malignant degree of the mesenchymal component in the recurrent neoplasm, sarcomatous transformation may occur. Hence, a long term clinical and radiographical follow-up is essential due to its transformation into ameloblastic fibrosarcoma.

[Primordial odontogenic tumor in children]. / Pervichnaya odontogennaya opukhol' u detei.

Primordial odontogenic tumor (POT) is a rare benign mixed epithelial and mesenchymal odontogenic tumor that has been included as a new nosological form in the latest classification of the World Health Organization (WHO 2017). The first two clinical cases of POT treatment in children in Russia are presented. A comprehensive examination and surgical treatment of POT were carried out. The diagnosis was confirmed morphologically. OBJECTIVE: To inform maxillofacial surgeons and dentists about the clinical, radiological and morphological features of POT on the example of clinical experience and literature data.

Decompression of Odontogenic Keratocysts Before Enucleation Reduces the Rate of Recurrence.

BACKGROUND: The purpose of this study was to determine if 2-stage conservative treatment (decompression followed by enucleation) of odontogenic keratocysts reduced the rate of recurrence compared to single-stage conservative treatment. METHODS: This was a pooled cohort study composed of both patients identified at our institution and cases reported in the literature. The primary predictor variable was treatment modality (single versus 2-staged conservative treatment). The outcome variable was the overall recurrence rate. RESULTS: A total of 684 patient cases were included in the final pooled sample. The mean age was 39.80years (range: 7-80 years). The mandible (76.8%) was the most common site, with the rest of the lesions manifesting in the maxilla (23.0%). A total of 58.1% of the lesions were unilocular, whereas the remaining 41.9% were multilocular. Two-stage conservative treatment demonstrated a lower recurrence rate (14.5% versus 22.1%; P = 0.029). CONCLUSIONS: Decompression is known to reduce lesion size and optimize the favorability of conservative treatment. Furthermore, we propose that 2-staged treatment may also be associated with a lower recurrence rate.

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.

Granular cell odontogenic tumor: report of a rare case and a review of literature.

BACKGROUND: Granular cell odontogenic tumor (GCOT) is a rare neoplasm with about 45 cases reported in the literature. It usually occurs in the posterior mandible of middle-aged women. CASE PRESENTATION: We report a case of asymptomatic GCOT in the posterior mandible of a 28 years old female and provide a literature review of GCOT cases. Some unusual features such as root resorption, displacement of inferior tooth canal, and multilocular appearance were considerable in this case. CONCLUSIONS: Complete surgical excision of the lesion was beneficial for the patient.

Central granular cell odontogenic tumor of mandible in a child.

Central granular cell odontogenic tumor (CGCOT) is sporadic benign odontogenic tumor and it especially occurs in women older than 50 years of age. Radiologically it manifests as unilocular to the multilocular radiolucency with sometimes mixed densities. Histopathology displays sheets and islands of large eosinophilic cells with abundant granular cytoplasm, however few cases exhibit inadequate epithelium, thus creating a diagnostic confusion. Though, resection is advocated by some surgeons, however because of the non-aggressive biological behaviour, enucleation or curettage is the treatment of choice for this lesion. Till now only 39 cases have been reported in the past six decades. We are reporting the first case of CGCOT occurring in the youngest age of eleven-year-old patient with massive size of 11 × 7 × 6 cm. This would add CGCOT as a differential diagnosis in the bony lesions of younger individuals. In addition, the importance of immunohistochemistry studies in cases with scarce odontogenic epithelium and the potential role of Carnoy's solution in the management of this rare tumor in this age group was emphasized.

Dentinogenic Ghost Cell Tumor associated with Odontoma: A unique Histopathological Entity and its Surgical Management.

We report a rare case of dentinogenic ghost cell tumor (DGCT) associated with complex composite odontoma in a 17 years male affecting the posterior segment of the mandible. On radiographic examination, there was a well-defined multilocular radiolucency surrounding the radio opaque mass with respect to 44, 45 and 46. Histopathologically it showed ameloblastomatous proliferation with dentin like areas and ghost cells. It was associated with tooth like structures consisting of dentin, cementum and pulp like areas. DGCT with odontoma is extremely rare with only two cases being reported in literature till date. The management with its rare occurrence is discussed here.

[Reconstruction of the mandible after loss of the middle segment due to an odontogenic myxoma]. / Reconstructie van de mandibula na verlies van het middensegment door een odontogeen myxoom.

In an 18-year-old boy, the middle segment of the mandible was removed because of a locally aggressive tumour. The reconstruction became infected and was lost, resulting in 2 separately-moving mandible parts and oral disability. For the second reconstruction, skeletal fixation with osteosynthesis plates, dental fixation with a stabilization frame and intermaxillary fixation were used. Preparation for returning the jaws to their original position was facilitated by three-dimensional simulation software. After a successful second reconstruction, an implant-supported removable bridge was eventually placed.

Primordial Odontogenic Tumor of Anterior Maxilla in a Young Male: A Case Report and an Updated Review of Literature.

Primordial odontogenic tumor (POT) is a recently designated benign mixed epithelial and mesenchymal odontogenic tumor and only sixteen cases have been reported worldwide till now. Here we report an another case of POT in a 14-year old boy who presented with an asymptomatic buccal enlargement in the right maxillary region for past 4-5 months. A well-defined, unilocular, radiolucent lesion with impacted teeth was observed radiographically. A detailed account of clinico-radiographical and histolological differential diagnoses along with an updated literature review has been presented.

Thirteen Synchronous Multifocal Calcifying Epithelial Odontogenic Tumors (CEOT): Case Report and Review of the Literature.

BACKGROUND: Calcifying epithelial odontogenic tumor (CEOT, Pindborg tumor) is a rare, benign, locally aggressive neoplasm of the jaws that accounts for approximately 1% of all odontogenic tumors. It was first defined by Pindborg in 1955 and has been reported approximately 350 times in the literature; 7 reported multiple (up to 4) synchronous lesions. MATERIALS AND METHODS: We report an individual with the largest number of CEOTs reported to date and provide a literature review of multifocal CEOT cases. RESULTS: A 30-year-old male presented to the Oral and Maxillofacial Surgery Department of the West Virginia University School of Dentistry (WVU SoD) to extract multiple impacted teeth previous to construction of a complete denture. A pantograph showed 15 impacted teeth, almost all associated with well-demarcated cyst-like radiolucencies, some with small, ill-defined radiopaque flecks. Microscopically, the lesions showed sheets and strands of polygonal epithelial cells with eosinophilic cytoplasm. Spread throughout the epithelium and connective tissue were small, spherical, amorphous, pale purplish calcifications. Each lesion was similar and consistent with a diagnosis of CEOT. CONCLUSION: We report a patient with 13 independent CEOTs scattered throughout all quadrants. This case represents the largest number of Pindborg tumors or any other type of odontogenic tumor yet reported in a single individual.