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The objective of this study is to present an uncommon case of adenomatoid odontogenic tumor (AOT) with an impacted maxillary canine, initially mimicking a follicular cyst. AOT is a rare odontogenic tumor, accounting for approximately 1% to 9% of all odontogenic tumors. It primarily occurs in the maxilla and is often associated with an unerupted permanent tooth. Follicular cysts, also known as dentigerous cysts, are benign odontogenic cysts that encase the crown of an unerupted or impacted tooth. We describe a case of AOT occurring in a 14-year-old male. Both the follicular cyst and adenomatoid odontogenic tumor (AOT) can exhibit similar clinical and radiographic presentations. It is crucial to accurately differentiate between the two to ensure appropriate treatment and prognosis. [ABSTRACT FROM AUTHOR]

Adenoid ameloblastoma is a newly recognized epithelial odontogenic tumor. Herein, we present the case of a 24-year-old male patient who exhibited swelling in the anterior region and right hemi-mandible. Computed tomography demonstrated the presence of a hypodense osteolytic lesion associated with an impacted tooth. Based on the clinical hypotheses of the dentigerous cyst, odontogenic keratocyst, and ameloblastoma, an incisional biopsy was performed, and the diagnosis of ameloblastoma was rendered. A surgical resection of the tumor was performed. Histopathological examination of the specimen revealed typical areas of ameloblastoma associated with ductiform structures and cell proliferation in a solid storiform pattern, features resembling those found in adenomatoid odontogenic tumor. Based on these findings, the diagnosis of adenoid ameloblastoma was rendered. The accurate diagnosis of this locally infiltrative tumor is essential due to its similarity to other odontogenic neoplasms. [ABSTRACT FROM AUTHOR]

The objective of this study was to review the clinical, radiologic, and histologic aspects of cases of dentigerous cysts metamorphosing into different entities along with comments on the molecular factors involved in the process. A series comprising 8 cases of dentigerous cysts was performed retrospectively along with a comprehensive literature review. Two cases of dentigerous cyst converting into mucous metaplasia were included, out of which one case was transformed into ameloblastoma after a year. The other two cases were reported as acanthomatous and unicystic ameloblastoma, whereas four transitioned to AOT. The complete review of cases and literature with molecular profiling concluded that the lining of dentigerous cysts has the potential for transforming into benign odontogenic tumors. Therefore, a careful clinical and histopathological examination is crucial for the correct diagnosis. Also, a thorough molecular understanding of the cystic lining is required. [ABSTRACT FROM AUTHOR]

The objective of this study is to present an uncommon case of adenomatoid odontogenic tumor (AOT) with an impacted maxillary canine, initially mimicking a follicular cyst. AOT is a rare odontogenic tumor, accounting for approximately 1% to 9% of all odontogenic tumors. It primarily occurs in the maxilla and is often associated with an unerupted permanent tooth. Follicular cysts, also known as dentigerous cysts, are benign odontogenic cysts that encase the crown of an unerupted or impacted tooth. We describe a case of AOT occurring in a 14-year-old male. Both the follicular cyst and adenomatoid odontogenic tumor (AOT) can exhibit similar clinical and radiographic presentations. It is crucial to accurately differentiate between the two to ensure appropriate treatment and prognosis.

Myotonic dystrophy, also referred myotonic muscular dystrophy, is an autosomal dominant, slowly progressive, multisystem disease characterized by skeletal muscle weakness, wasting, and myotonia. A hybrid tumor of odontogenic apparatus is a lesion showing combined histopathological characteristics of two or more previously recognized odontogenic tumors and/or cysts of different categories. We, therefore, report a case of hybrid tumor (adenomatoid odontogenic tumor associated with calcifying cystic odontogenic tumor) in a myotonic dystrophic patient. [ABSTRACT FROM AUTHOR]

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Introduction: Odontogenic tumors encompass a heterogeneous group of lesions that range from hamartomatous lesions to malignancy. Considerable variation in histologic presentation can mislead their accurate diagnosis and categorization. Ameloblastoma is generally well understood and is easy to diagnose but there has been a constant change in the classification systems ever since Broca classified odontogenic tumors in the year 1867. Over the years, it has been modified by the World Health Organization with many additions and omissions. This dynamic change is based on the result and conclusions of molecular and genetic studies with the last modification in 2017. Case Report: We present two cases of females aged 32 and 60 years who reported with facial swellings, revealed the presence of distinct histopathological findings and were diagnosed as ameloblastoma with dentinoid or adenoid ameloblastoma. Literature search revealed dearth of distinct forms of ameloblastoma that show the formation of duct like structures and dentinoid. Conclusion: It is interesting to highlight such cases as the biological behavior is still unexplored due to paucity of relevant studies and follow up of patients. Understanding the pathogenesis and the histopathological characteristics of the newer entities will enable the prompt diagnosis, treatment plan and expanding the spectrum of the lesions. [ABSTRACT FROM AUTHOR]

Background: Adenoid ameloblastoma (AdAM) is a frequently recurrent tumor that shows hybrid histological features of both ameloblastoma and adenomatoid odontogenic tumor (AOT). AdAM is expected to be classified as a new subtype of ameloblastoma in the next revision of the World Health Organization (WHO) odontogenic tumor classification. However, whether AdAM is a histologic variant of ameloblastoma or AOT remains unclear. To establish a new category, genetic evidence indicating the tumor category is necessary. Methods: We present a case of a 23-year-old Japanese woman with AdAM who underwent genetic/DNA analysis for ameloblastoma-related mutation using immunohistochemical staining, Sanger sequencing, and next-generation sequencing (NGS) analyses with reliable clinicopathological evidence. Results: Immunohistochemical expression of BRAF p.V600E was diffusely positive for both ameloblastoma- and AOT-like components. Sanger sequencing and NGS analyses showed missense mutations in BRAF p.V600E (c.1799T > A), a gene that is commonly altered in ameloblastomas but not in KRAS, another gene associated with AOT. Conclusion: This case report is the first to provide genetic evidence on the ameloblastomatous origin of AdAM with a BRAF p.V600E mutation. A larger series of AdAM groups' molecular testing is needed to aptly classify them and prognosticate the best treatment. [ABSTRACT FROM AUTHOR]

Background: This study aimed to investigate the differentiation of ameloblastic‐like cells and the nature of the secreted eosinophilic materials in adenomatoid odontogenic tumors. Methods: We studied histological and immunohistochemical characteristics of 20 cases using: cytokeratins 14 and 19, amelogenin, collagen I, laminin, vimentin, and CD34. Results: Rosette cells differentiated into ameloblastic‐like cells positioned face‐to‐face, displaying collagen I‐positive material between them. Epithelial cells of the rosettes can differentiate into ameloblastic‐like cells. This phenomenon probably occurs due to an induction phenomenon between these cells. The secretion of collagen I is probably a brief event. Amelogenin‐positive areas were interspersed by epithelial cells in the lace‐like areas, outside the rosettes and distant from the ameloblastic‐like cells. Conclusions: There are at least two types of eosinophilic material in different areas within the tumor, one in the rosette and solid areas and another in lace‐like areas. The secreted eosinophilic material in the rosettes and solid areas is probably a product of well‐differentiated ameloblastic‐like cells. It is positive for collagen I and negative for amelogenin, whereas some eosinophilic materials in the lace‐like areas are positive for amelogenin. We hypothesize that the latter eosinophilic material could be a product of odontogenic cuboidal epithelial or intermediate stratum‐like epithelial cells. [ABSTRACT FROM AUTHOR]

Background: The process of odontogenesis is complex involving epithelial-mesenchymal interactions, along with the molecular signalling pathways triggering the initiating process. The triggering factors and cells precisely involved in the pathogenesis of odontogenic cysts and tumors are unknown. There is a vast array of biomarkers used to stain different sites, thereby helpful in diagnosing and evaluating the prognosis of these cysts and tumors. In the following study, Anti Apoptotic survivin expression patterns were assessed quantitatively in 48 samples (12 each) of Reduced Enamel Epithelium, Adenomatoid Odontogenic Tumor, Odontogenic Keratocyst and Ameloblastoma. Aim: The Aim of this study is to assess the anti-apoptotic survivin expression in Reduced Enamel Epithelium, Adenomatoid odontogenic tumour, Odontogenic Keratocyst and Ameloblastoma. Materials and Methods: The present study is carried out with 12 samples in each group. Routine hematoxylin and eosin staining was performed for confirmatory diagnosis. Later Immunohistochemistry was performed using survivin antibody. Survivin protein expression was analyzed using the parameters like location, intensity, percentage of cells positivity with survivin protein and extent of staining. With the help of Olympus BX 43 microscope, with ProgRes microscope camera, the 48 slides obtained were examined. The region of interest was selected in each slide and number of cells positively stained was counted. Data was analyzed using SPSS software version 23. Descriptive for scale data, results were analysed by using ANOVA with Chi-square test for intergroup comparison. Results: The results showed significant P value <0.05. Expression of survivin was highest in Ameloblastoma, followed by Odontogenic keratocyst, Adenomatoid odontogenic tumor, and Reduced Enamel Epithelium. Conclusion: Survivin was involved in the inhibition of apoptosis as well as the detailed understanding of the biological behaviour of odontogenic cysts and tumours, thereby increasing therapeutic approaches. [ABSTRACT FROM AUTHOR]

Adenomatoid odontogenic tumor (AOT) is a benign slow-growing, asymptomatic epithelial odontogenic neoplasm. This lesion has been known for its varied clinical and histoarchitectural patterns. Most AOTs occur intra-osseously in the anterior maxilla associated with the unerupted tooth. Clinically, AOT is sometimes misdiagnosed as an odontogenic cyst. Although enucleation and curettage for AOT is the most common treatment modality, accurate histopathological diagnosis is essential to avoid unnecessary extensive surgery. Here, we present a rare challenging case of an extrafollicular cystic adenomatoid odontogenic tumor occurring in the body of the mandible in a 23-year-old female patient, which was diagnosed clinically and radiographically as glandular odontogenic cyst. The diagnosis of adenomatoid odontogenic tumor was confirmed through histopathological examination.

Adenomatoid Odontogenic Tumor (AOT) is an uncommon odontogenic benign epithelial neoplasm. It accounts for about 2.2–7.1 % of all odontogenic tumors. Two clinicopathological intraosseous variants (Follicular and Extrafollicular) and one extraosseous variant (Peripheral) have been described. The follicular variant is considered the predominant form (about 75 % of all reported cases). An unerupted maxillary canine is the tooth most commonly associated with a follicular AOT (FAOT). The extrafollicular type (EAOT) is infrequent and is not related to an unerupted tooth. EAOT variant do not show distinctive clinical radiological features. Recurrence is very rare, which justify its conservative treatment. In this paper, we report a case of a 15-year-old female who had EOAT in the anterior mandible. Additionally, an update review on the etiology, age and gender distribution, tumor localization, clinic and radiological presentation, symptoms, histopathology/immunohistochemistry (IHC), diagnosis, treatment and recurrence of this condition is presented. [ABSTRACT FROM AUTHOR]

Background: The occurrence of hybrid odontogenic lesions with two or more morphologically distinct components is a rare phenomenon and poses a diagnostic challenge. We aimed to study the clinical, radiological, and pathological features and behavior of hybrid odontogenic lesions, to enhance awareness about these rare lesions. Method: Hematoxylin and Eosin slides of hybrid odontogenic lesions diagnosed between January 01, 2012 and December 31, 2020, were reviewed. Demographic and radiological information were obtained from the patient's medical records. Results: 8 cases were diagnosed with a mean age of 19.1 years and male to female ratio of 1:1.7. Involvement of mandible was more common (n = 5) as compared to maxilla (n = 3). All patients presented with swelling for an average of 9.75 months (3–25 months) duration. Bleeding, loose teeth, pain and facial asymmetry were reported in 5,3, 3, and 2 cases, respectively. Radiologically, 7 cases were well demarcated, 75% cases (n = 6) were radiolucent, and average radiological size was 4.8 cm. All patients were managed with surgery alone. 5 cases (62.5%) underwent enucleation and curettage, while local excision, en-block resection and segmental mandibulectomy were performed in 1 case each. Histologically, ossifying fibroma/cemento-ossifyiong fibroma were the most lesion, occurring in 5 cases (62%), followed by giant cell granuloma like lesions (GCG) i.e., central and peripheral giant cell granuloma (n = 3), Adenomatoid Odontogenic tumor (AOT) (n = 2), and DC (n = 2), ameloblastic fibroma (AF) (n = 1), Ameloblastoma (n = 1), calcifying odontogenic cyst (COC) (n = 1), and complex odontoma (n = 1). No evidence of recurrence was noted after 4–99 months of surgery (mean: 32.9) in cases with available data (n = 7). Long-term complaints included facial asymmetry (n = 2) and pain (n = 1). Conclusion: Most hybrid odontogenic lesions affect young females in the second decade of life and commonly show COF and OF as hybrid components. A conservative approach to management appears adequate.

The odontogenic tissue surrounding an impacted tooth is a harbinger of a wide variety of odontogenic cysts and tumors. As these hamartomatous and neoplastic aberrations can occur at any stage of odontogenesis, therefore combined features of odontogenic tumors with epithelial and mesenchymal components can develop within the odontogenic cysts. One of the odontogenic cysts is the dentigerous cyst (DC) which is not only the most common developmental odontogenic cyst arising from the pericoronal tissue but is of great clinical significance owing to its neoplastic potential and association with other rare odontogenic tumors. We herein describe an unusual case of adenomatoid odontogenic tumor which developed secondary to a DC in a 22-year-old female and created a diagnostic conundrum. The approach to such a case, differential diagnosis, and review of the literature is also presented.

An adenomatoid odontogenic tumor (AOT) is an uncommon benign tumor of the oral cavity commonly found in the maxillary anterior region and is associated with impacted canines in young females. It rarely occurs in the mandibular region with no impacted or missing teeth. A 21-year-old female reported to the clinic with swelling on the right side of the face for the past six months with no history of pain. Radiographic features such as unilocular radiolucency with thinning of the cortical borders and considerable buccal cortical expansion, as well as some evidence of radiopaque specks were noted. Histopathological examination revealed cells with hyperchromatic nuclei, rosette-like structures with focal areas of calcified mass, and concentric rings resembling Liesegang rings, suggestive of AOT. The tumor was treated surgically by enucleation and cauterization. Although follicular type is a common variant, the tumor presented in this case was of extrafollicular type noted in the mandibular canine and premolar regions of a young female patient with no related impacted tooth.

Adenomatoid odontogenic tumor (AOT) is a benign slow-growing, asymptomatic epithelial odontogenic neoplasm. This lesion has been known for its varied clinical and histoarchitectural patterns. Most AOTs occur intra-osseously in the anterior maxilla associated with the unerupted tooth. Clinically, AOT is sometimes misdiagnosed as an odontogenic cyst. Although enucleation and curettage for AOT is the most common treatment modality, accurate histopathological diagnosis is essential to avoid unnecessary extensive surgery. Here, we present a rare challenging case of an extrafollicular cystic adenomatoid odontogenic tumor occurring in the body of the mandible in a 23-year-old female patient, which was diagnosed clinically and radiographically as glandular odontogenic cyst. The diagnosis of adenomatoid odontogenic tumor was confirmed through histopathological examination. [ABSTRACT FROM AUTHOR]

This study aims to report two distinct cases of adenomatoid odontogenic tumor (AOT) with contrasting presentations. The first case involved a 12-year-old female patient with the presence of AOT in the posterior mandible extending into the mandibular ramus up till the coronoid process which is a rare reported site for AOT and the second case is of a 19-year-old male patient with the tumor in the anterior maxilla with an impacted central incisor. Both cases were successfully managed via surgical enucleation and are under regular follow-ups without any complication. The contrasting nature of the two clinical presentations with the same pathologic entity is reported here. Though AOT has been rarely reported extending into the mandibular ramus, a maxillofacial surgeon should be more circumspect of this pathology while dealing with benign swellings of the mandibular posterior region. [ABSTRACT FROM AUTHOR]

Adenomatoid Odontogenic Tumor (AOT) is one of the rare tumor of oral cavity which predominantely occurs in the anterior maxilla. This is a case report of a Mandibular AOT successfully treated by complete removal of the tumor and transplantation of canine has been done in the same time and splinted with arch bar. The least chance of recurrence reported in literature in the treatment of AOT favours the surgical plan which applied in the treatment of this lesion in the case reported. This case report highlights the advantages of auto transplantation in reconstruction of the lost teeth along with surgical removal of the tumor and the extraction of retained deciduous teeth. [ABSTRACT FROM AUTHOR]

This article aimed to collectively present the demographic, clinical, radiographic and histopathological features as well as the treatment performed along with its outcome for all the cases of adenoid ameloblastoma with dentinoid (AAD) reported in scientific literature till date. Ameloblastoma and adenomatoid odontogenic tumours are the most common odontogenic neoplasms. However, AAD, a hybrid variant of the two lesions, is found to be extremely rare. The lesion comprises of characteristic histopathological features of ameloblastoma and adenomatoid odontogenic tumour and shares certain clinical characteristics with either of the entities. AAD may be considered to be present at the more aggressive end of spectrum of benign odontogenic neoplasms. Owing to the frequent tendency of the lesions to be underdiagnosed, careful histopathological screening of submitted biopsies is warranted. With the increase in number of reported cases in the recent years, it is likely to be included as a separate entity in the upcoming World Health Organization classification. [ABSTRACT FROM AUTHOR]

Adenomatoid odontogenic tumor (AOT) is an infrequent odontogenic tumor that typically occurs in adolescent females, usually in the anterior maxilla. There is a controversy about it being a tumor or a hamartoma. It presents clinically as a slowly progressive entity that shows a good prognosis with conservative surgical management. It shows three clinicopathological types: follicular, extrafollicular, and peripheral. This article describes a follicular variety of AOT. An 18-year-old female presented with diffuse intraoral swelling in the maxillary anterior region. An intraoral periapical radiograph (IOPA) revealed a single large pericoronal radiolucency related to the impacted right maxillary lateral incisor. Histopathological evaluation confirmed the diagnosis, and the tumor was treated surgically by enucleation., Competing Interests: Human subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: In compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services info: All authors have declared that no financial support was received from any organization for the submitted work. Financial relationships: All authors have declared that they have no financial relationships at present or within the previous three years with any organizations that might have an interest in the submitted work. Other relationships: All authors have declared that there are no other relationships or activities that could appear to have influenced the submitted work., (Copyright © 2024, Dangore-Khasbage et al.)

There are rarely hybrid neoplasms composed of two or more distinct histological types; a hybrid odontogenic tumor with a combination of the calcifying epithelial odontogenic tumor (CEOT) is present, adenomatoid odontogenic tumor (AOT) and calcifying odontogenic cyst (COC), located in the anterior mandible region from a 15-year-old male patient causing mild mobility of anterior mandible incisors with the labial and lingual expansion of the mandible cortical plates and obvious facial asymmetry. The tumor was surgically removed through an en-bloc resection approach. Microscopic examination revealed an atypical hybrid odontogenic tumor, with features of the three entities previously mentioned. Several cases of hybrid neoplasms have been reported in the literature. However, there is no evidence about the incidence of a hybrid odontogenic tumor with histologic features of CEOT combined with two other entities. In combination with COC, this is a unique presentation of a hybrid tumor not previously documented.

Adenomatoid Odontogenic Tumor (AOT) accounts for 3 % of all odontogenic tumors. It has been classified by WHO as an odontogenic tumor of purely epithelial origin. The current study attempts to establish the origin of the tumor along with detailed histopathological and clinicoradiographic analysis of 43 cases of AOT. Forty-three cases were reviewed from the departmental archives for demographic data, radiographic features and histological features. Further, histopathological slides were stained with Picrosirius Red (PSR) and observed under polarised light. A majority of the cases were seen in the anterior jaws (76.7 %), and were less than 3 cms (76.7 %) in greatest dimension. Equal number of cases were of follicular and extra-follicular location while one was peripheral. Predominantly solid histological pattern was noted in 53.5 %. Varied sub-patterns were observed with most cases exhibiting solid nodules and strands of tumor cells. Few cases showed melanin pigmentation. Over a third of cases (37.2 %) showed dentigerous cyst like areas and one case each showed features of ossifying fibroma and focal cemento-osseous dysplasia. Tumor droplets, hyaline rings within duct-like structures, dentinoid material and osteodentin showed reddish yellow birefringence when observed under polarised microscopy post PSR staining. This study highlights the diverse histopathological variation of AOT with evidence to reclassify it as a mixed odontogenic tumor based on the polarising microscopic findings with PSR staining. • Picrosirius red staining along with polarised microscopy has been used to highlight the mesenchymal component within AOT. • The study challenges conventional understanding of AOT by proposing a mixed odontogenic origin. • A detailed clinical, radiographic and pathologic analysis has been done, aiding in better understanding of the topic. [ABSTRACT FROM AUTHOR]

Background: Adenomatoid odontogenic tumor (AOT) is an uncommon odontogenic tumor that originates Tom odontogenic epithelium. It is predominantly found in young women, particularly in the maxilla in association with an unerupted permanent tooth. Case description: This is a case report of an AOT in a 60-year-old male with an asymptomatic mandibular lesion that radiographically presented as a mixed lesion. Conclusions: Enucleation was performed and the defect was treated with bone grafting and platelet-rich librin with no recurrence at follow-up. [ABSTRACT FROM AUTHOR]

We present two cases of AOT, the first case concerns a 23‐year‐old patient with an AOT located in the maxilla and the second case involves a 37‐year‐old patient presenting an AOT with mandibular localization. [ABSTRACT FROM AUTHOR]

The odontogenic tissue surrounding an impacted tooth is a harbinger of a wide variety of odontogenic cysts and tumors. As these hamartomatous and neoplastic aberrations can occur at any stage of odontogenesis, therefore combined features of odontogenic tumors with epithelial and mesenchymal components can develop within the odontogenic cysts. One of the odontogenic cysts is the dentigerous cyst (DC) which is not only the most common developmental odontogenic cyst arising from the pericoronal tissue but is of great clinical significance owing to its neoplastic potential and association with other rare odontogenic tumors. We herein describe an unusual case of adenomatoid odontogenic tumor which developed secondary to a DC in a 22-year-old female and created a diagnostic conundrum. The approach to such a case, differential diagnosis, and review of the literature is also presented. [ABSTRACT FROM AUTHOR]

Objectives: Calcifying odontogenic cysts (COC) and adenomatoid odontogenic tumors (AOT) have similar radiographic findings. We examined the radiographic and computed tomography (CT) images of patients histologically diagnosed with COC or AOT and identified their characteristic findings. Methods: The subjects included 12 patients histologically diagnosed with COC or AOT (one female and five males per group), who underwent CT at our hospital between Nov 1998 and Jun 2019. The location of the lesion, impacted tooth, bone expansion, root resorption, tooth migration, calcified body, and presence or absence of a high-intensity zone in the marginal area of the lesion were examined. Results: In patients with COC, five patients with COC exhibited bone expansion toward the buccal side. The lesion encompassing the crown was attached to the cement-enamel junction and contained a radiopaque lesion with a calcified body. In 6 patients with COC, irregularly shaped calcified bodies were observed with small tooth-like structures. In patients with AOT, all six patients with AOT exhibited bone expansion toward the buccal and lingual sides. The lesion encompasses a part of the tooth root or the entire tooth. Punctate calcification was observed within the lesion and the marginal area in three patients, and a high-intensity zone was observed in the marginal area of the lesion in two patients. Conclusion: We report imaging findings that may be characteristic of COC and AOT, suggesting that CT findings may be useful for differentiating between COC and AOT. [ABSTRACT FROM AUTHOR]

Abstract We present two cases of AOT, the first case concerns a 23‐year‐old patient with an AOT located in the maxilla and the second case involves a 37‐year‐old patient presenting an AOT with mandibular localization.

Adenomatoid Odontogenic Tumor (AOT) is a benign, non-invasive tumor with slow but progressive growth, mainly affecting younger patients, predominantly females. It is more often located in maxilla, involving an unerupted or erupted tooth, mostly canine. There are three variants, namely follicular, extra-follicular and peripheral. Permanent cuspids account for 60% of all follicular and 89% of all extra-follicular AOT. This article discusses a unique case of extra-follicular AOT in 9 year old male patient associated with partially erupted maxillary central incisor.

Adenomatoid odontogenic tumor (AOT) is benign tumor of the oral cavity characterized by its slow growth accounting to 3%–7% of the odontogenic tumors. AOTs mostly occur in the maxillary anterior region and most often than not associated with impacted anterior teeth. It affects the younger age group, especially below 20 years and is commonly seen in females. It occurs as two main variants – (a) central/intraosseous which is more common and (b) peripheral which is rare. The location of the lesion, its association with the impacted tooth, is the basis for the classification of AOT. The intraosseous type may be related to unerupted tooth (follicular variant) or may not be related to unerupted tooth (extra-follicular variant). Radiologically, AOT presents predominantly as a unilocular cystic lesion enclosing the unerupted tooth. The lesion presents rarely with a cystic component. Radiopacities on the intraoral periapical radiograph are seen as discrete foci having a flocculent pattern within radiolucency even with the presence of minimal calcified deposits. AOT being benign in nature is enucleated with least chances of recurrence. Here, we present a rare case of extra-follicular variant of adenomatoid odontogenic tumor in a 19-year-old female patient.

Purpose: To perform a comprehensive and systematic critical appraisal of the genetic alterations reported to be present in adenomatoid odontogenic tumor (AOT) compared to ameloblastoma (AM), to aid in the understanding in their development and different behavior.Methods: An electronic search was conducted in PubMed, Scopus, and Web of Science during March 2021. Eligibility criteria included publications on humans which included genetic analysis of AOT or AM.Results: A total of 43 articles reporting 59 AOTs and 680 AMs were included. Different genomic techniques were used, including whole-exome sequencing, direct sequencing, targeted next-generation sequencing panels and TaqMan allele-specific qPCR. Somatic mutations affecting KRAS were identified in 75.9% of all AOTs, mainly G12V; whereas a 71% of the AMs harbored BRAF mutations, mainly V600E.Conclusions: The available genetic data reports that AOTs and AM harbor somatic mutations in well-known oncogenes, being KRAS G12V/R and BRAFV600E mutations the most common, respectively. The relatively high frequency of ameloblastoma compared to other odontogenic tumors, such as AOT, has facilitated the performance of different sequencing techniques, allowing the discovery of different mutational signatures. On the contrary, the low frequency of AOTs is an important limitation for this. The number of studies that have a assessed the genetic landscape of AOT is still very limited, not providing enough evidence to draw a conclusion regarding the relationship between the genomic alterations and its clinical behavior. Thus, the presence of other mutational signatures with clinical impact, co-occurring with background KRAS mutations or in wild-type KRAS cases, cannot be ruled out. Since BRAF and RAS are in the same MAPK pathway, it is interesting that ameloblastomas, frequently associated with BRAFV600E mutation have aggressive clinical behavior, but in contrast, AOTs, frequently associated with RAS mutations have indolent behavior. Functional studies might be required to solve this question.

Adenomatoid odontogenic tumor (AOT) is a rare, asymptomatic, slow-growing benign tumor that can be divided into three variants: follicular, extrafollicular, and peripheral. By treating AOT using an enucleation and curettage approach, recurrence can be avoided. We report a case of a 24-year-old female who presented with a lump in the right mandibular premolar area along with diastema between displaced teeth #43 and #44 and was diagnosed with extrafollicular AOT. The patient was managed with enucleation-curettage surgery without additional bone graft procedure along with routine follow-up. A successful outcome without recurrence was achieved, and diastema closure with repositioning of the displaced teeth did not require orthodontic treatment. AOT should be managed via enucleation and curettage to obtain successful outcomes without recurrence. Spontaneous bone regeneration following enucleation can be achieved without guided bone regeneration. Also, diastema closure and repositioning of displaced teeth can occur without orthodontic interventions through physiologic drift.

Adenomatoid odontogenic tumor (AOT) is a rare tumor comprising about 2.2% to 7.1% of all odontogenic tumors. It is a neoplasm of odontogenic origin having slow-growth rate, noninvasive and benign in nature. Here, we report a case of a young female patient who presented with an aggressive AOT in maxilla involving the impacted third molar. Usually, the treatment for AOT comprises of enucleation and curettage, whereas due to the fast growth rate and aggressiveness of the lesion, subtotal maxillectomy was performed in this case. The authors have described about the clinical, radiographic, histopathological and surgical aspect of the case. [ABSTRACT FROM AUTHOR]

Abstract Background Adenomatoid odontogenic tumor constitutes an uncommon benign odontogenic tumor which is well-known as “two-thirds tumor” (two-thirds of adenomatoid tumors occur in the maxilla, two-thirds occur in young females, two-thirds of the cases are associated with un-erupted teeth and two-thirds of the affected teeth are canines). Larger part of these present as intra - osseous tumors while peripheral counterparts are extremely rare. Peripheral adenomatoid odontogenic tumor is a unique entity which generally presents as a slow growing gingival swelling with minimal bone involvement. This often leads to its erroneous diagnosis as a simple gingival lesion and the real diagnosis is only revealed after its microscopic evaluation. It exhibits a marked predilection for maxillary gingiva of incisor region and most commonly affects the younger females. Case presentation We report a case of 25 years old female patient of African ethnicity who presented with a 2 × 2 cm mass attached to the left mandibular gingiva in cuspid- bicuspid region which is an unusual site for AOT. It was accompanied by slight bone loss. With the differential diagnosis of gingival epulis and peripheral ossifying fibroma; surgical excision was performed. The light microscopic examination of the specimen aided the final diagnosis of Adenomatoid odontogenic tumor with the histopathological features identical to its intra osseous counterpart. Conclusion In this case, the tumor is present on the mandibular gingiva in a 25 years old patient which is an atypical location and age for this tumor; also, the only individual case reported in an African patient. Only, a meager number of peripheral adenomatoid odontogenic tumor cases have been logged so far with majority of them occurring in maxillary gingiva. Furthermore, an ambiguity still prevails regarding its true origin and possible biological course. Hence, reporting of similar cases should be encouraged to facilitate the better understanding of its varied epidemiological details and clinical presentation.

An impacted tooth is the one which fails to erupt into its anatomic position due to various factors such as malposition, lack of space, or other impediments. The prevalence of impacted tooth varies with geographic locations and has been estimated to be between 8% and 38%. An impacted tooth can be a nidus for dental caries, infection, destruction of adjacent teeth, periodontal disease, and even oral and maxillofacial cysts or tumors. Therefore, careful evaluation of the history and the clinical and radiographic findings and knowledge about the various maxillofacial cysts and tumors help a clinician to arrive at early diagnosis and render proper treatment to a patient. The recent advancements made in the field of radiology, especially the cone-beam computed tomography (CBCT), is a boon to dentistry. The three-dimensional imaging has made CBCT the preferential imaging modality for the evaluation of unerupted/impacted or supernumerary teeth and also in the evaluation of odontogenic and nonodontogenic cysts/tumors of the jaws. The varied radiographic appearances of cysts/tumors in the maxillofacial region help in precise differentiation of the pathology and prompt the clinicians to arrive at an accurate early diagnosis. This article discusses three case reports of maxillofacial cysts/tumors associated with an impacted tooth where radiology plays an important role in the diagnosis of the lesions.

Adenamatoid odontogenic tumor (AOT) is an odontogenic tumor with a prevalence of 2.2–7.1%. AOT is a benign, noninvasive, and progressive lesion which is also known as “a two third tumor.” As the name suggests the tumor occurs in the maxilla in two third of cases. It occurs in young patients in two third of cases and associated with missing or unerupted teeth in two third of cases. Two third cases are associated with the maxillary canine. Characterized by slow growing, gradually enlarging, painless swelling associated with missing teeth. We report a case of a male patient of age 22 years, with characteristic findings. AOT resembles different odontogenic cysts and tumors which may include dentigerous cyst, globulomaxillary cyst, ameloblastoma, and other entities, hence must be well differentiated. Conservative surgical enucleation is the treatment of choice. Recurrence rate for AOT is 0.2%. Prognosis is excellent when completely removed in toto.

Hybrid odontogenic tumors are sporadic, where the distinctive areas of more than one odontogenic tumor tissue type have been reported. The occurrence of adenomatoid odontogenic tumor (AOT) with calcifying epithelial odontogenic tumor (CEOT) like areas histologically simulating salivary gland pathology is an unusual finding that has not been previously reported in the literature. We report the case of a 32-year-old female presenting with slow-growing firm swelling, radiographically as a pear-shaped radiolucent lesion in the interdental region of maxillary incisors. Histologically, the tissue showed nests and anastomosing strands of the bland cuboidal to squamoid epithelial cells showing nuclear pleomorphism, hyperchromatism, and abundant cytoplasm with prominent intercellular bridges focally. Multiple basophilic calcifications, amyloid-like material, duct-like formation, and mucinous spillage are seen. Tumor cells showed immunopositivity for CK 7, CK 19, CK 8/18 and low Ki67, p63, and immunonegativity for S100 suggesting of a hybrid lesion of CEOT with AOT. [ABSTRACT FROM AUTHOR]

Objective: Integrins function to bind cells to extracellular matrix in tissues, which triggers downstream signaling cascades that are important in cell survival, proliferation, cytokine activation, and cytoskeleton reorganization. These processes also play significant roles in neoplasms. This work aimed to investigate the pattern of expression of FAK, paxillin, and PI3K in ameloblastoma and adenomatoid odontogenic tumor (AOT). Materials and methods: Immunohistochemistry was used to study FAK, paxillin, and PI3K in 45 ameloblastomas (32 conventional, 12 unicystic, and 1 peripheral types), 7 AOTs, and two developing human teeth. Results: Weak expression of FAK was seen in all AOT cases, while ameloblastoma had varying expression patterns, mostly strong to weak. The pattern of expression of paxillin and PI3K was relatively similar in both tumor types. In the dental germ, FAK and paxillin stained all the enamel organ components, while PI3K stained strongly the inner enamel epithelium. Stromal expression of FAK was not found to be useful in differentiating between tumors or tumor classes. Conclusion: The expression of the proteins in the enamel organ suggests that their signaling may be important in odontogenesis. While some ameloblastomas strongly expressed FAK, all cases of AOT had weak signals suggesting low presence and phosphorylating activity of FAK in the latter. Clinical relevance: A subset of FAK-positive ameloblastoma (as well as their malignant or metastasizing counterparts) which may have relatively aggressive behavior may be candidates for drug targeting of FAK as an additional management option. [ABSTRACT FROM AUTHOR]

A 12-year girl presented with a firm well‑defined swelling in her left maxilla. Radiologically, there was a well-circumscribed, unilocular radiolucency with dimension about 35 × 30 mm, and related to the crown of the unerupted permanent left canine. The computed tomography (CT) reveled an extensive lesion elevating the floor of maxillary sinus. Then, it was performed aspirations, incisional biopsy, decompression, and enucleation of the lesion. The diagnosis was compatible with adenomatoid odontogenic tumor. The treatment of this lesion is the entire removal. So, the patient was evaluated periodically, completing 3 years of following up. New CT were taken at 5 and 22 months post-operative, showing the complete regression of the cavity where the lesion was. [ABSTRACT FROM AUTHOR]

Ameloblastoma is one of the most common types of oral odontogenic tumors. As per literature, ameloblastoma mostly occurs in the mandible butthe maxillary ameloblastoma has a more aggressive behavior due to anatomical features. Also, unicystic ameloblastoma may have lower recurrent rate. In this case report, we present a 60-year-old male patient with a history of unicystic ameloblastoma, which the intraluminal adenomatoid odontogenic tumor excisional biopsy surgery was performed but the patient didn’t follow the treatment completely, and after two years he came back with swelling of the right upper alveolar ridge. After the second surgery, the histopathologic report revealed a mixed plexiform-follicular ameloblastoma recurrence and it seemedthat previous surgery was not sufficient and more radical treatment is needed for the lesion. Keywords: Ameloblastoma; Maxilla; Adenomatoid odontogenic tumor.

Adenomatoid odontogenic tumor (AOT) is a benign epithelial tumor that shows duct-like structures. AOT is usually associated with a permanent tooth, and maxillary canines are the most frequently affected. However, an association between intraosseous AOTs and unerupted primary teeth is extremely rare. Herein, we report an unusual case of an AOT associated with an unerupted primary tooth, which we initially attempted to preserve. A 4.5-year-old female with an AOT associated with the primary maxillary left first molar initially underwent surgical enucleation of the lesion, which preserved the tooth without postoperative eruption, followed by curettage and extraction during a second surgery. The maxillary left first premolar, which was positioned mesially to the primary tooth, showed eruption tendencies at 5 years after the initial surgery. The present case demonstrates that AOT should be included in the differential diagnosis for cystic lesions in children, since AOTs can develop around their unerupted primary teeth. Furthermore, while treating AOT, the decision of whether to preserve the associated primary or permanent tooth should be given careful consideration. [ABSTRACT FROM AUTHOR]

Adenomatoid odontogenic tumor is a non-aggressive slow growing epithelial odontogenic tumor with varied clinical and microscopic features. This neoplasm rarely appears as a completely cystic lesion histopathologically; therefore, oral pathologists should be aware of the various histopathologic types of this lesion. The main objective of this case report is to present a rare challenging case of maxillary extrafollicular cystic adenomatoid odontogenic tumor affecting a 28-year-old male.

Adenomatoid odontogenic tumor (AOT) is a rare noninvasive benign tumor of the jaw that originates in the second decade of the life from the odontogenic epithelium without mesenchyme. Radiological examination is an essential component for the diagnosis of benign tumors, the radiographic modality used to acquire the image was speculated to have an effect on the perception of radiopacities, particularly for the cases with minimal intralesional calcifications. Therefore, the capability of radiographic modality in detecting the intralesional calcification was critical for diagnosis of AOT. Cone beam computed tomography (CBCT) provides precise imaging of bony structures with minimal calcification without superimposition and distortion. In this case report, we are reporting a case of AOT of maxilla with characteristic CBCT appearance of AOT of maxilla.

Adenomatoid odontogenic tumor (AOT) is benign tumor of the oral cavity characterized by its slow growth accounting to 3%–7% of the odontogenic tumors. AOTs mostly occur in the maxillary anterior region and most often than not associated with impacted anterior teeth. It affects the younger age group, especially below 20 years and is commonly seen in females. It occurs as two main variants – (a) central/intraosseous which is more common and (b) peripheral which is rare. The location of the lesion, its association with the impacted tooth, is the basis for the classification of AOT. The intraosseous type may be related to unerupted tooth (follicular variant) or may not be related to unerupted tooth (extra-follicular variant). Radiologically, AOT presents predominantly as a unilocular cystic lesion enclosing the unerupted tooth. The lesion presents rarely with a cystic component. Radiopacities on the intraoral periapical radiograph are seen as discrete foci having a flocculent pattern within radiolucency even with the presence of minimal calcified deposits. AOT being benign in nature is enucleated with least chances of recurrence. Here, we present a rare case of extra-follicular variant of adenomatoid odontogenic tumor in a 19-year-old female patient. [ABSTRACT FROM AUTHOR]

Adenomatoid odontogenic tumor (AOT) is a relatively rare, benign, hamartomatous, and cystic odontogenic neoplasm that was first described more than a century ago. It accounts for 2-7% of all odontogenic tumors. The lesion still continues to intrigue experts with its varied histomorphology and controversies regarding its development. The present article describes a case of cystic AOT with an unusual histomorphology associated with an impacted 43 in a 15-year-old male. [ABSTRACT FROM AUTHOR]

Claudins are integral to the structure and function of tight junctions. Altered claudin expression has been shown to affect disease behavior and patient prognosis in various neoplasms. The objectives of this study were to analyze the claudin-1, -4 and -7 expression in odontogenic tumors and characterize their expression pattern in distinct tumor cell types in relation to the recurrence potential. Sixty-nine cases of odontogenic tumors, including 43 ameloblastomas (AM), 17 adenomatoid odontogenic tumors (AOT), 6 ameloblastic fibromas (AF) and 3 ameloblastic carcinomas (AC) were investigated for claudin-1, -4 and -7 expression immunohistochemically. The staining was analyzed semi-quantitatively and categorized into 4 levels, based on the percentage of positively stained neoplastic epithelial cells. Claudin-1 was expressed in all AOT and AF cases, whereas most AC (66.7%) showed no expression. The claudin-1 staining was moderate-to-intense in the odontogenic epithelium of AF. In contrast, its staining of ameloblast-like cells and stellate reticulum-like cells in AM was weak. Claudin-7 expression was noted in all tumor types studied, while the expression of claudin-4 was limited and mainly localized in the squamous differentiated cells of AM and AC. AM showed significantly higher claudin-4, but lower claudin-7 expression than AOT. In addition, AC showed diminished claudin-1 immunoreactivity, compared to AOT. Low claudin-1 expression in AM was significantly associated with the increased clinical recurrence. The loss of claudin-1 may underlie the locally invasive nature of AM. [ABSTRACT FROM AUTHOR]

Two cases reports of adolescents from both sexes are described, they presented edemas in the left side of the maxillary with dental reabsorption and in the anterior portion of the maxillary, respectively. The histopathology revealed an intraosseous folicular variety of the adenomatoid odontogenic tumor. The surgical treatment used was an enucleation with satisfactory results. [ABSTRACT FROM AUTHOR]