Benign Odontogenic Tumor Research Articles

Histopathological spectrum of primordial odontogenic tumor with co-existing dentigerous cyst: 1st reported case of the world with a proposed ‘updated diagnostic criteria’

BackgroundPOT is a relatively newly described benign odontogenic tumor with very few cases registered to date. We present the 1st case of Primordial odontogenic tumor (POT) from Sub-Saharan Africa with unique clinicopathological features; also, this is the first case to report POT’s existence as a Hybrid Odontogenic lesion (HOL), with a pertinent review of the literature.Case presentationThis was a 17-year-old patient who presented with slow-growing, painless posterior mandibular swelling. The imaging revealed a well-defined, unilocular, expansile, lytic lesion with internal calcific foci surrounding an impacted #36, indicating a calcifying odontogenic cyst. The incisional biopsy revealed the presence of POT. The tumor was excised along with the involved tooth.ConclusionPOT is predominantly a non-aggressive and mostly affects the pediatric population. Hence, clinicians must be updated on all the aspects of this tumor to diagnose it appropriately and avoid any undue over—or under-treatment.

Central odontogenic fibroma: retrospective study of six cases with variable histopathologic features using 2022 WHO classification

BackgroundCentral odontogenic fibroma (COF) is a rare benign odontogenic tumor with a wide range of histopathologic features. We evaluated COF diagnosed in our institute with 16 years’ experience using 2022 WHO classification.MethodsOur archives were reviewed and cases diagnosed as COF were selected. Clinical, radiographic and microscopic features were tabulated and analyzed.ResultsOf 13,736 specimens, six cases (0.04%) of COF were discovered. Patients ranged in age from 14 to 44 years. There were two males and four females. Maxilla and mandible were affected equally. Five cases showed radiolucent appearance (4 unilocular, 1 multilocular) and one case showed a mixed radiolucent-radiopaque pattern. Histopathologically, three cases were conventional type (2 epithelium-rich, 1 epithelium-poor). Two cases were the hybrid COF with central giant cell granuloma (CGCG) and one of which was also associated with secondary aneurysmal bone cyst (ABC). The last case with a mixed radiolucent-radiopaque pattern was the ossifying subtype.ConclusionOur results demonstrated that COF is a rare odontogenic tumor and exhibits diverse radiographic and microscopic appearances. The triphasic tumor consisting of the COF, CGCG and ABC, is reported here for the first time, while the ossifying subtype is considered the tenth case reported in the English-language literature. Oral and maxillofacial pathologists and other healthcare personnel must be aware of this rare odontogenic tumor and its variants to achieve the definite diagnosis.Clinical trial numberNot applicable.

Squamous Cell Carcinoma Arising in a Squamous Odontogenic Tumor of the Maxilla: Case Report and Review of the Literature.

Squamous odontogenic tumor (SOT) is an exceedingly rare, benign epithelial odontogenic tumor showing squamous differentiation. It is composed of variably sized and shaped islands of cytologically bland, mature squamous epithelium within a fibrous stroma. In this report, we present a rare transformation of a squamous odontogenic tumor (SOT) of the maxilla into a well-differentiated squamous cell carcinoma (SCC) with involvement of the pterygoid plates. To the best of our knowledge, only two cases of malignant transformation of SOT has been reported in the literature. Herein, we seek to report this extremely rare occurrence to raise awareness of oral and maxillofacial surgeons and pathologists of this unusual, but serious event and perform a literature review of squamous odontogenic tumors.

A Challenging Case Of Giant Maxillary Ameloblastoma: Navigating Rarity And Complexity In Diagnosis And Treatment

Background: Ameloblastoma is a rare, aggressive, benign odontogenic tumor that most commonly occurs in the jaw. Maxillary occurrences are rarely reported. Case Presentation: We report the case of a 61-year-old male presenting with right-sided unilateral nasal obstruction, characterized by a mass occupying the entire right nasal fossa upon anterior rhinoscopy. A computed tomography (CT) scan, combined with magnetic resonance imaging (MRI), revealed a locally advanced mass centered on the right maxillary sinus. The patient underwent wide excision of the mass, and histopathological analysis confirmed the diagnosis of ameloblastoma. Discussion: Naso-sinus ameloblastomas are rare, benign, and slow-growing tumors, accounting for approximately 0.11% of nasal and sinus tumors. More common in men, they typically present between the ages of 20 and 30. Symptoms include nasal obstruction and epistaxis, with occasional facial swelling or headaches. These tumors often originate in the posterior maxillary region and extend into the maxillary sinuses and nasal cavity. Diagnosis is facilitated by imaging, where the tumors appear as solid, radio-opaque masses. Radical surgical resection is the preferred treatment, as it is essential for preventing recurrence. The prognosis is generally favorable, with no documented cases of malignant transformation or metastasis, and recurrences are rare when appropriate surgical management is performed. Conclusion: Medical imaging plays a crucial role in diagnosis and allows for the evaluation of tumor dissemination and anatomical involvement. The treatment of maxillary ameloblastoma typically involves surgical excision, with the complexity of management often linked to the extent of tumor spread.

Mandibular Ameloblastoma: A Reported Case

Ameloblastoma is a benign aggressive odontogenic tumor which requires early diagnosis and appropriate treatment. It commonly affects the mandible and radical surgery is the gold standard treatment. We report the case of a patient with ameloblastoma in extremely advanced phase affecting the retromolar trigone who was treated with concomitant chemoradiation therapy. Mandibular ameloblastoma is not well documented in the literature. It is usually diagnosed at the later stage when optimal surgery cannot be performed. This case study aimed to demonstrate that radiation therapy is a real therapeutic alternative in the treatment of advanced and inoperable forms of ameloblastoma.

MMP13 Expression and Activity Suggest Its Role in Bone Resorption in Ameloblastomas.

Ameloblastoma is a locally destructive benign odontogenic tumor. While the neoplastic cells of conventional ameloblastoma can infiltrate the connective tissue and bone, in unicystic ameloblastoma the epithelium is encapsulated. The mechanisms driving ameloblastoma's bone resorption remains unclear. RNA sequencing (RNA-seq) was performed in a discovery cohort of conventional ameloblastoma, and pathway enrichment analysis was carried out. mRNA levels of MMP13, a gene associated with bone resorption, were assessed using RT-qPCR in a larger cohort of conventional ameloblastoma and in unicystic ameloblastoma. Zymogram gels and the immunoexpression profile of collagenase 3 (encoded by MMP13 gene) were evaluated as well. Enriched pathways related to bone mineralization and upregulation of MMP13 were observed in ameloblastomas. Collagenolytic activity of collagenase 3 was detected in the tumor lysates. Collagenase 3 immunopositivity was observed in ameloblastomatous epithelium infiltrating the fibrous capsule of unicystic ameloblastoma. At the tumor-bone interface, collagenase 3 expression was detected in stromal cells, osteoblasts, and osteocytes. The results indicate a potential involvement of MMP13 in ameloblastoma-related bone resorption and progression.

Ameloblastoma Cases in the Mandible: Experience from the Professor Hamady Traoré National Center of Odonto-Stomatology of Bamako

Introduction: Mandibular ameloblastoma is the most common benign odontogenic tumour in stomatological and maxillofacial surgery practice. The aim of this work was to study the epidemiological, anatomopathological and therapeutic aspects of mandibular ameloblastoma in our context. Methodology: It was a descriptive cross-sectional study over a 4-year period (January 2017 to December 2020) of cases of mandibular ameloblastoma confirmed by histology of the operative specimen in the stomatology and and maxillofacial surgery department of the CHU-CNOS in Bamako. Data were collected on a survey form, entered and analyzed using SPSS version 19.0 software. Results: There were 44-recorded cases. Males accounted for 61.36% of cases, with an M/F sex ratio of 1.6. The mean age was 33 years, with a standard deviation of 12 years. The 20-40 age bracket was in the majority (52.27%). In 79.55% of cases, patients consulted for swelling with facial deformity. Dental signs such as mobility, displacement or absence were found in 97.72% of cases. Hemi-mandibular location was the most frequent (45.45%). The main histological type was follicular ameloblastoma in 88.64% of cases. Radical surgery with disarticulation was performed in 47.73% of cases. Conclusion: Mandibular ameloblastoma is common in young adults. Radical surgery is the current trend. Reconstructive techniques are still limited in a context of underdevelopment.

Comprehensive insights into the understanding of hypoxia in ameloblastoma.

Hypoxia is characterized by a disparity between supply and demand of oxygen. The association between hypoxia and head and neck tumors is a topic of significant interest. Tumors frequently encounter areas with inadequate oxygen supply, resulting in a hypoxic microenvironment. Ameloblastoma is one of the most common benign odontogenic tumors of the maxillofacial region. It is a slow-growing but locally invasive tumor with a high recurrence rate. The literature has demonstrated the correlation between hypoxia and ameloblastoma, revealing a discernible link between the heightened expression of hypoxic markers in low oxygen conditions. This association is intricately tied to the tumoral potential for invasion, progression, and malignant transformation. Hypoxia profoundly influences the molecular and cellular landscape within ameloblastic lesions. The present review sheds light on the mechanisms, implications, and emerging perspectives in understanding this intriguing association to clarify the dynamic relationship between hypoxia and ameloblastoma.

Central dentinogenic ghost cell tumor of the maxilla: a case report with new imaging findings and review of the literature

A dentinogenic ghost cell tumor (DGCT) is a rare benign odontogenic tumor that commonly shows characteristics of solid proliferation and has a relatively high risk of recurrence after surgical treatment. We herein report a case of a central DGCT that occurred in the maxilla and resulted in bone expansion. This study highlights new imaging findings (particularly magnetic resonance imaging) along with histopathological observations. In addition, we conducted a review of the existing literature on this rare tumor. A 37-year-old man developed swelling around the right cheek. A benign odontogenic tumor such as ameloblastoma was suspected based on the imaging examination findings (including bone expansion and the internal characteristics of the tumor) on panoramic imaging, computed tomography, and magnetic resonance imaging. The lesion was surgically excised from the right maxilla. Postoperative histopathological examination led to a definitive diagnosis of central DGCT. The tumor comprised epithelial neoplastic islands, resembling ameloblastoma, inside tight fibroconnective tissue; masses of ghost cells and formation of dentin were also observed. We had suspected that the minute high-density region around the molars on the imaging examinations represented alveolar bone change; however, it represented dentin formation. This led to difficulty diagnosing the lesion. Although DGCT may present characteristic findings on imaging examinations, its occurrence is infrequent, and in some cases, the findings may include the presence or absence of an impacted tooth without obvious calcification. The present case suggests that we should consider the possibility of an odontogenic tumor with calcification when high-density structures are observed inside the lesion.

Ameloblastic fibro-odontoma with a congenitally missing mandibular second molar : a rare case report

Ameloblastic fibro-odontoma (AFO) is a rare benign mixed odontogenic tumor predominantly occurring in children. This tumor is often asymptomatic and associated with disturbances in tooth eruption. We present the case of a 7-year-old boy who exhibited delayed eruption of his left mandibular first molar, attributable to AFO with a congenitally missing mandibular second molar. Radiographs revealed a well-defined, ovoid, radiolucent lesion containing sparse calcified materials in the left posterior mandible. To the best of our knowledge, few reports have documented AFO in conjunction with the congenital absence of a permanent tooth. This case is discussed along with a review of relevant literature.

Compound composite odontome-A report of seven case series with brief review of literature.

Odontomes are the most common benign odontogenic tumors of the jaw which are usually slow-growing and non-aggressive. They are generally asymptomatic and diagnosed during routine radiographic investigations and are mostly associated with tooth eruption disturbances. The present case series report covers seven cases of odontomes discussing on clinical, radiographic, and histopathologic features (ground section and decalcification) along with a brief review of the literature.

Odontogenic myxoma: A dual affliction in maxilla and mandible - A case report

The odontogenic myxoma (OM) is a locally invasive benign odontogenic tumour of the jaws, thought to originate from the mesenchymal portion of the tooth germ. It commonly manifests in individuals during their second or third decade of life.Females experience a higher frequency of occurrences compared to males, and there is a greater inclination for odontogenic myxoma to affect the mandible rather than the maxilla. In most cases, OM manifests as an asymptomatic swelling. Due to its non-capsulated and aggressive nature, there is a notable high recurrence rate reported.Small myxomas are typically addressed through curettage, but it is crucial to conduct thorough follow-up examinations for a minimum of 5 years. The standard treatment for myxoma typically involves surgical resection. Our article highlights two instances of odontogenic myxoma occurring in separate locations. The emphasis is on summarizing distinctive clinical and radiological behaviour of these lesions manifesting in the maxilla and mandible.

Anti-MAPK Targeted Therapy for Ameloblastoma: Case Report with a Systematic Review.

Ameloblastoma, a benign yet aggressive odontogenic tumor known for its recurrence and the severe morbidity from radical surgeries, may benefit from advancements in targeted therapy. We present a case of a 15-year-old girl with ameloblastoma successfully treated with targeted therapy and review the literature with this question: Is anti-MAPK targeted therapy safe and effective for treating ameloblastoma? This systematic review was registered in PROSPERO, adhered to PRISMA guidelines, and searched multiple databases up to December 2023, identifying 13 relevant studies out of 647 records, covering 23 patients treated with MAPK inhibitor therapies. The results were promising as nearly all patients showed a positive treatment response, with four achieving complete radiological remission and others showing substantial reductions in primary, recurrent, and metastatic ameloblastoma sizes. Side effects were mostly mild to moderate. This study presents anti-MAPK therapy as a significant shift from invasive surgical treatments, potentially enhancing life quality and clinical outcomes by offering a less invasive yet effective treatment alternative. This approach could signify a breakthrough in managing this challenging tumor, emphasizing the need for further research into molecular-targeted therapies.

SURGICAL MANAGEMENT OF DESMOPLASTIC AMELOBLASTOMA: CONSERVATIVE OR RADICAL APPROACH

Ameloblastoma is a benign odontogenic tumor of epithelial origin. It is locally aggressive with unlimited growth capacity and a high potential for malignant transformation and metastasis. (1) Nowadays, there are two opposing approaches to the surgical management of ameloblastoma. The conservative approach consists of enucleation and/or curettage and the radical approach consists of bone resection with margins of at least 1cm (segmental or not). (2) In this work, we will present a case of desmoplastic ameloblastoma diagnosed in 2014. The patient had been benefiting from regular follow-up. However, a recurrence occurred in 2021. This paper delves into our experiences in managing desmoplastic ameloblastoma, initially employing a conservative approach in 2014 and later transitioning to a radical approach in 2021 in response to the recurrence.

The Curious Case of a 'Maverick' Cementoblastoma!

The Cementoblastoma is a benign ectomesenchymal odontogenic neoplasm of the jaw, arising from an exuberant production of cemental tissue continuous with the roots of teeth (usually mandibular posteriors), resulting in a calcified mass adherent with the root apices. A curious case of a large Cementoblastoma, the size and shape of a ping pong ball, is presented, the first of its kind ever reported to involve the internal / medial / lingual aspect of the mandible, and presenting with unusual and deviant features. The Cementoblastoma is usually associated with certain inherent and pathognomonic features, making it rather straightforward to identify and diagnose. Strikingly uncharacteristic clinical, radiographic and histologic attributes presented in this case, made this 'out of the ordinary' and 'Maverick'-like Cementoblastoma, arduous to diagnose, nevertheless, making it an interesting, informative and curious Case Study. This report has brought to light for the first time, several new facets of the Cementoblastoma.

Low Detection of High-risk Human Papilloma Virus in Individuals with Ameloblastoma in a Tertiary Hospital in Lagos, Nigeria.

Ameloblastoma is a benign but aggressive epithelial odontogenic neoplasm of unknown etiology. The role of human papilloma virus (HPV) in the etiology of oral squamous cell carcinoma has prompted the investigation of HPV as an etiologic factor in ameloblastoma. This study aimed to determine the frequency of high-risk (HR) HPV in conventional ameloblastoma and the clinical parameters associated with infection. The study was approved by the ethical review boards of the institution. DNA was extracted from fresh tissue collected 750 μL of DNA/RNA Shield (Zymo Research, United States) using Invitrogen PureLink Viral RNA/DNA Mini Kit (Invitrogen, USA). The extracted DNA was assayed for the detection of 14 HR HPV types (16, 18, 31, 33, 35, 39, 45, 51, 52, 56, 58, 59, 66, and 68) using Anyplex™ II HPV HR Detection kit (Cat. No. HP7E00X) (Seegene Inc., Republic of Korea) on CFX-96TM Real-Time Polymerase Chain Reaction (PCR) System (Bio-Rad). Data on gender, age of patient, site of lesion, clinicohistological types of ameloblastoma and history of smoking, alcohol consumption, and practice of oral sex were collected. Data analysis was performed using analysis program SPSS version 25 and statistical significance was set at P < 0.05. Two cases of conventional ameloblastoma were positive with HPV and none of the ameloblastic carcinoma cases were positive. The HPV 16 serotype was observed in both cases. While 5 of the cases had a history of alcohol consumption, none of these cases were positive for HPV serotype. HPV 16 positivity was detected in two cases of conventional ameloblastomas and none in ameloblastic carcinoma using real-time PCR. There was no effect of exposure to smoking, alcohol consumption, and practice of oral sex and HPV in the etiology of ameloblastoma. Data available are suggestive of a limited role of HPV in the etiology of ameloblastoma.

Ameloblastic fibroma of mandible- A case report

Ameloblastic fibromas are uncommon mixed benign odontogenic tumors with an overall prevalence of less than 5%. Asymptomatic uniloular lesions can be effectively managed with curettage, multilocular lesions require meticulous enucleation to avoid the risk of relapse. We report a case successful management of an asymptomatic multilocular ameloblastic fibroma with 2 years of follow up.

Follicular Ameloblastoma of Maxillary: A Case Report

Introduction: Ameloblastoma is a benign odontogenic tumor that appears in the mandible and maxilla. Ameloblastoma has slow growth that takes several years for symptoms to appear, but ameloblastoma in the maxilla has a more aggressive clinical course compared to the mandible. The lack of initial symptoms leading to diagnosis at an advanced disease when the tumour has already extended beyond maxilla. The cancellous structure of the maxillary bone makes it easier for tumors to spread to the nasal cavity, paranasal sinuses, orbitals, parapharyngeal tissue and skull base. This article reports a case of resection of maxillary ameloblastoma. Case Report: A 63 year old female patient came with complaints of a lump on her right cheek since 2 years back that gradually increased in size. Since 2 months ago, pus and blood have been coming out of the lump. Extra oral examination revealed a well-defined mass with a solid and hard consistency, there were no ulcers or fistulas. Intraorally, a mass measuring 5x5x3cm was found in the region of teeth 12 to 15 which extended to the buccal-palatal area. History of allergies and weight loss was denied. Supporting examinations including panoramic radiography, PA chest radiography, CT-Scan, FNAB, and biopsy resulted in a diagnosis of ameloblastoma. Treatment was carried out by resection of the right maxillary tumor mass under general anesthesia. Discussion: In this case, the patient's clinical examination showed a hard and large palpable swelling in the right maxilla area which extended from the region of tooth 12 to tooth 15 which caused facial asymmetry and no tooth mobility was found. This hard and painless swelling takes about 2 years before the patient experiences symptoms of pus and blood discharge. The post-maxillectomy defect in this case was covered with an intraoral prosthesis in the form of a post-surgical obturator made of acrylic resin. The obturator functions to hold the surgical packing, and prevent food or dirt contamination in the defect area which can cause infection and slow healing. The use of a prosthesis also helps restore swallowing and speech function to the patient. Conlussion: In principle, the treatment for ameloblastoma is resection of the involved bone, as in this case the action taken is a maxillectomy. Maxillectomy can result in facial and oral cavity deformities characterised by facial disfigurement and alterations in oral functionality.Therefore, maxillary reconstruction is needed to treat maxillary defects after surgical procedures involving the loss of part or all of the maxilla.

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

BackgroundAmeloblastoma (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 presentationWe 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.ConclusionsChildren 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.

A Complex Odontoma in a Paediatric Patient with Hypodontia: A Case Report and Review of the Literature

The complex odontoma is a benign odontogenic tumor of the jaw, consisting of a disorganised mass of dental tissue with unknown aetiology. They are often asymptomatic and usually identified incidentally upon radiographic examination. However, they have the potential to cause displacement and resorption of adjacent teeth as well as affecting tooth eruption, therefore early identification is important. We report a case of a 12 year old male who was referred to our department with an incidental finding of a complex odontoma in the mandible.