Adenomatoid odontogenic tumor, an uncommon tumor

Here we report a case of adenomatoid odontogenic tumor (AOT) in the maxilla in a young girl aged 14 years and its surgical management. We also review the literature and variations in the nomenclature and classifications of this interesting tumor. The review of literature gives an interesting picture regarding terminologies in the past and dilemma in classifying this tumor. The introduction of the name adenomatoid odontogenic tumour has resulted in the simpler and fruitful surgical management like enucleation and curettage with no reports of recurrences. In the past, similar lesion with the terminology like adeno ameloblastoma has resulted in unnecessary mutilating surgery. The conflicting views whether the lesion is being neoplasm or an anomalous hamartomatous growth is also being discussed.     

Two adenomatoid odontogenic tumours of the maxilla: A case report

An adenomatoid odontogenic tumour (AOT) is a well-recognised benign tumour that enlarges slowly. We report here the rare occurrence of two separate tumours in the anterior maxillary region that have not been documented previously. They were associated with unusual findings such as relatively large size, a supernumerary tooth, rapid growth with cortical perforation, and multilocular appearance with root resorption on plain radiograph.     

Adenomatoid odontogenic tumor mimicking a periapical (radicular) cyst: a case report

The adenomatoid odontogenic tumor (AOT) is a benign (hamartomatous), noninvasive lesion with slow but progressive growth. A rare subvariant of the extrafollicular type of AOT may mimic periapical disease radiographically. We report on a 15-year-old girl with a periapical radiolucent lesion affecting teeth 7 and 8 initially suspicious of periapical disease, although clinical findings seemed to indicate a nonendodontic lesion. An exploratory surgical approach was chosen, and the final diagnosis of the removed noncystic tissue mass was microscopically confirmed to be an AOT.     

Proliferating cell nuclear antigen (PCNA) and p53 protein expression in ameloblastoma and adenomatoid odontogenic tumor

In this study, proliferating cell nuclear antigen (PCNA) and p53 protein expressions were analyzed in 16 cases of ameloblastoma and 8 cases of adenomatoid odontogenic tumor (AOT). The cases of ameloblastoma consisted of solid type tumors and histologic arrangements of different subtypes were observed. In some specimens, more than one histologic subtype was identified in the same lesion, and each tumor was categorized according to the predominant cell pattern. The odontogenic tumors were grouped as follows: follicular ameloblastoma (n=7), plexiform ameloblastoma (n=4), acanthomatous + follicular ameloblastoma (n=3), basal cell ameloblastoma (n=2), adenomatoid odontogenic tumor (n=8). PCNA immunohistochemical expression revealed stronger quantitative labeling index for the follicular ameloblastoma, while for p53 protein the strongest quantitative labeling index was detected in the plexiform type. Nevertheless, statistical analysis using ANOVA and Tukey's test did not detect significant differences (p>0.05) among the histologic subtypes of ameloblastoma. The findings of this study suggest that the different histologic patterns of ameloblastoma did not show a direct correlation with their clinical behavior and consequently with the prognosis of the cases. The results also indicated that the ameloblastoma has greater proliferative potential than the AOT, which can contribute to explain its more aggressive and invasive characteristics.     

Adenomatoid odontogenic tumour: A case report

Adenomatoid odontogenic tumour (AOT) is a rare benign odontogenic tumour characterized by a progressively slow growing pattern and symptomless behavior. The differential diagnosis between AOT and other odontogenic tumours, such as ameloblastoma, should be well conducted in order to avoid extensive ablative surgery. This report presents an unusual case of an 11-year-old male patient who referred to the oral surgeon due to a significant painless gingival swelling in the anterior mandible. A panoramic X-ray revealed a round radiolucid image of an intraosseous lesion with well defined boards and related to the left lateral incisor and left canine. The Computerized Tomography was performed and the sagittal sections revealed a tooth image in contact with the inferior board of the tumour. Additionally, the coronal sections showed the presence of a tooth inside the lesion. Several calcifying nodules could be distinguished within the cystic area. The clinical diagnostic hypothesis was of calcifying epithelium odontogenic tumour but the histological sections were consistent with AOT. The tumour was enucleated under local anesthesia. After one year follow-up there were no signs of reoccurrence. With respect to the distinguishing tumour enlargement and localization in the lower jaw, the reported case is an uncommon example of AOT.     

Odontogenic gingival epithelial hamartoma with a pleomorphic histological appearance: a case report and review of the literature

Odontogenic gingival epithelial hamartoma (OGEH) is a rare odontogenic epitheliallesion. Distinguishing OGEH from peripheral ameloblastoma can pose a differential diagnostic challenge. We report a case of OGEH affecting the lower gingiva in a 78-year-old male patient. The lesion presented as a large, painless, pedunculated mass with a pleomorphic histological appearance. A diagnosis of OGEH was made after the exclusion of peripheral ameloblastoma and peripheral adenomatoid odontogenic tumour. Previous studies have reported differences in sex and age between patients with OGEH and peripheral ameloblastoma; this suggests that peripheral ameloblastoma is not derived from OGEH. Histological findings suggest that the lesions are independent entities.     

Adenomatoid odontogenic tumor--a rare cause of jaw swelling

Adenomatoid odontogenic tumor (AOT) is an uncommon tumor of odontogenic origin, characterized histologically by the formation of ductlike structures with amyloid-like deposits. Histogenesis of AOT is still uncertain and it is often considered as a hamartomatous lesion rather than a true neoplasm. AOT has a benign behavior and conservative surgical enucleation or curettage is sufficient. We report a case of AOT in a 15-year-old female who presented with left-sided jaw swelling with tooth resorption. Histopathology revealed intraosseus follicular variant of AOT. A brief review of literature is also discussed.     

Atypical plexiform ameloblastoma with dentinoid: adenoid ameloblastoma with dentinoid

In this study, we report a tumor that resembled previously reported uncommon tumors histologically similar to ameloblastoma or adenomatoid odontogenic tumor (AOT), showing the formation of hard tissue. We evaluated the histological characteristics by reviewing the literature. The patient was a 19-year old male. The lesion was located from the canine to third molar in the right mandible and was unicystic with a comparatively clear demarcation. The tumor tissue was cystic overall, showing multiple formation of small and large cysts. The tumor tissue resembled a variant form of plexiform ameloblastoma. Formation of dentin and dentinoid was observed in the tumor stroma, whereas formation of enamel was not observed. Very few cases of a variant form of ameloblastoma that shows formation of dentinoid have been reported, and the histological picture in this study closely resembled previously reported "adenoid ameloblastoma with dentinoid".     

Adenomatoid odontogenic tumour: review of the literature and an analysis of 33 cases from South Africa

The adenomatoid odontogenic tumour (AOT) is a benign lesion of odontogenic origin. It is a slow growing tumour that results in a painless expansion of the jaws. This is a retrospective review of the demographic, clinical and radiographic features of AOTs diagnosed in a black South African population over 20 years. Of the 746 odontogenic tumours diagnosed, 4% were AOTs. The patients’ ages ranged from 9 to 37 years with a mean age of 15 years. The highest incidence was in the second decade of life (85%). The female to male ratio was 5.6:1. The maxilla was more commonly affected than the mandible in a ratio of 1.5:1. The sizes of the lesions ranged from 2 to 7 cm, with 60% involving an entire quadrant. All were of the central follicular type and appeared as well-demarcated radiolucent lesions. The canine was the most common impacted tooth. The treatment of choice was enucleation of the lesion, with no recurrences being reported.     

Adenomatoid Odontogenic Tumor (AOT) Arising from a Dentigerous Cyst: Literature Review and Report of a Case

Adenomatoid odontogenic tumor (AOT) is an uncommon benign tumor of odontogenic origin composed of odontogenic epithelium in a variety of histopathological patterns. These lesions are usually solid but are occasionally cystic. AOT has been reported to occur in association with odontogenic cysts. Very few cases have been described that arise in association with a dentigerous cyst. A systematic search of the English-language medical literature in PubMed and Medline search (keywords adenomatoid odontogenic tumor, dentigerous cyst) data bases revealed only ten such cases.The present case is very unique, exceptional and first of this kind that occurred in the posterior mandible associated with an impacted lower first premolar in a 19-year old female patient. The intraosseous follicular AOT, as this case reported here, frequently resemble dentigerous cysts. There is an uncertainty whether the lining of an associated cyst represents a true dentigerous cyst, cystic change within an AOT or may represent a distinct entity. We believe that this case represents an odontogenic cyst with neoplastic change. Hence, in such cases, the final diagnosis should be made on the basis of histological examination of extensive tissue sampling of entire excised tissue specimen. Further studies to determine whether the AOT derived from an odontogenic cyst could represent a distinct variant are to be done.     

Extrafollicular adenomatoid odontogenic tumor: a case report

The adenomatoid odontogenic tumor (AOT) is a benign, non-invasive lesion with a slow but progressive growth. This tumor has three variants: follicular, extrafollicular, and peripheral. The extrafollicular type is an intraosseous lesion that is not associated with unerupted teeth. Radiographically, it appears as a well-defined, unilocular, round or ovoid radiolucency with multiple, small, irregular radiopaque foci. Microscopically, it is characterized by nodules of cuboidal or columnar epithelial cells that form nests, rosette or duct-like structures (with minimal stromal connective tissue), and acellular calcifications. This article presents the case of a 35-year-old man with a lesion on the anterior mandible. Clinical and radiographic features indicate that this lesion is an extrafollicular variant of AOT.     

Variants of the adenomatoid odontogenic tumor with a note on tumor origin

Three rare variants of the adenomatoid odontogenic tumor (AOT) are described. A follicular AOT associated with an impacted and displaced 28, an extra-follicular variant mimicking a radicular cyst around the apex of 23 and a peripheral (epulis-like) variant exhibiting a periodontal bone defect palatal to 21. On reappraisal of the origin and pathogenesis of the AOT, it would seem that this tumor or hamartomatous lesion is derived from odontogenic epithelium of the dental lamina complex or its remnants.     

Follicular adenomatoid odontogenic tumor: immunohistochemical study

Adenomatoid odontogenic tumor (AOT) is an uncommon benign odontogenic lesion that affects young patients, with female predominance, mainly in second decade, showing a radiolucent unilocular image associated with an unerupted tooth, usually a canine. In spite of previous and confusing denominations, such as adenoameloblastoma or adenomatoid ameloblastic tumor, AOT is a benign tumor with a very low rate of recurrence, that show a peculiar morphological picture (basaloid appearance with glandular-like structures, calcifying areas, and amiloid-like material) that allow its histopathological recognition. We present a clinicopathological analysis of a case of follicular AOT affecting the mandible in a 9 years-old female patient associated with unerupted lower left canine. Immunohistochemical study showed some data previously unrecognised. All cellular types that composed AOT showed nuclear positivity for p63 indicating a basal characterization in the different cellular components. According to its benign character and low potential for recurrence, AOT revealed a scant proliferative activity (2-3% nuclei showed Ki-67 positivity) limited to some epithelial nodules (AE1-3 +) of fusiform appearance. Absence of reactivity for hormonal receptors (RE and RPg) excluded a possible hormonodependence in AOT that could explain the observed female predominance.     

Rare presentation of adenomatoid odontogenic tumor in a pediatric patient: a case report

The adenomatoid odontogenic tumor (AOT) is a painless benign tumor with slow growth, usually asymptomatic. It has three variants: follicular, extrafollicular, and peripheral. In the follicular type, the tumor is associated with an impacted tooth, and maxillary canines are the most frequently affected. Association with primary teeth is very rare. Treatment consists essentially in a total lesion enucleation. The objective of this paper is to present the clinical case of a 7-year-old female patient with an adenomatoid odontogenic tumor associated with the crown of the left lower deciduous canine (73), dislodging it to the mandibular base and consequently shifting and also impacting the permanent lower canine (33). The lesion was treated with careful enucleation, preserving the permanent canine, which then had its eruption path released favoring its migration to an ideal position.     

The adenomatoid odontogenic tumour: an update of selected issues

The aim of this update was to present the recent notable progress within remaining questions relating to the adenomatoid odontogenic tumour (AOT). Selected issues that were studied included the following: (i) AOT history and terminology, (ii) the so‐called peripheral AOT, (iii) AOT and the gubernaculum dentis and (iv) the so‐called adenomatoid odontogenic cyst (AOC). The earliest irrefutable European case of AOT was described in 1915 by Harbitz as ‘cystic adamantoma’. Recently, Ide et al. have traced two Japanese cases with irrefutable proof described by Nakayama in 1903. The so‐called peripheral (gingival) variant of AOT seems to cover a dual pathogenesis, both an ‘erupted intraosseous’ and an ‘extraosseous’ (gingival) one. In 1992, we theorized that the generally unnoticed gubernaculum dentis (cord and canal) seems to be involved in the development of AOT. Ide et al. have concluded that the dental lamina in the gubernacular cord seems to be an embryonic source of the vast majority of AOTs. The suggestion by Marx and Stern to change the nomenclature of AOT to adenomatoid odontogenic cyst (AOC) is critically discussed. The present authors agree on the background of the work of several groups of researchers and WHO/IARC classifications that the biology of the follicular variant of AOT is already fully explained and does not make room for any change in diagnostic terms. Further, there is no reason to change terminology in this case where improvements or conditions to better clinical management are not an issue.     

Ameloblastic Carcinoma

Ameloblastic carcinoma (AC) is a rare aggressive malignant epithelial odontogenic tumor of the maxillofacial skeleton with a distinct predilection in the mandible. It may appear de novo or originate from a pre-existing ameloblastoma or odontogenic cyst. It exhibits cytological features of ameloblastoma and carcinoma. It may present as a cystic lesion with benign clinical features or as a large tissue mass with ulceration, significant bone resorption and tooth mobility. The clinical course of ameloblastic carcinoma is typically aggressive, with extensive local destruction. Direct extension of the tumour, lymph node involvement and metastasis to various sites has been reported. Wide local excision is the treatment of choice. Regional lymph node dissection should be considered and performed selectively. Radiotherapy and chemotherapy have limited role in the treatment of ameloblastic carcinomas. Close periodic reassessment of the patient is mandatory.     

Odontogenic tumors: a review of 319 cases in a Nigerian teaching hospital

OBJECTIVE: This study sought to determine the relative frequency of odontogenic tumors in a Nigerian population and to compare these data with previous reports. STUDY DESIGN: Records of patients seen at the Lagos University Teaching Hospital between January 1980 and December 2003, with histologic diagnosis of odontogenic tumors (based on World Health Organisation classification, 1992), were analyzed. RESULTS: Odontogenic tumors constituted 9.6% of all the biopsies of oral and jaw lesions seen within the period under study. Three hundred and eight (96.6%) were intraosseous, and 11 (3.4%) were peripheral (peripheral odontogenic fibroma=7; peripheral myxoma=3; peripheral ameloblastoma=1). The mean age of patients was 29.9+/-15.6 years (range, 4-85 years). Among these cases, 96.6% of the tumors were benign and 3.4% were malignant. Ameloblastoma with predilection for the mandible was the most frequent odontogenic tumor (63%), followed by adenomatoid odontogenic tumor (AOT) (7.5%), myxoma (6.5%), calcifying epithelial odontogenic cyst (5.3%), and odontogenic fibroma (5.3%). More cases of malignant odontogenic tumors were seen than cases of calcifying epithelial odontogenic tumor and odontomas. The mean ages of patients with AOT, ameloblastic fibroma, and odontoma were significantly lower than those with ameloblastoma ( P<.05). No significant difference was found between the mean ages of patients with benign odontogenic tumors and those with malignant odontogenic tumors ( P=.058). CONCLUSIONS: Odontogenic tumors, especially ameloblastoma, are not considered rare among Nigerians, whereas odontoma, regarded as the most frequent odontogenic tumor in North and South America, is rare.     

Neoplastic potential of odontogenic cysts

Odontogenic cysts and tumors are distinct entities and quite a common occurrence in the jaw bones. The lining of odontogenic cysts shows a potential for neoplastic transformation to non odontogenic malignancies like squamous cell carcinoma and mucoepidermoid carcinoma, and odontogenic tumors like ameloblastoma and adenoamatoid odontogenic tumor (AOT). AOT is a benign, epithelial odontogenic tumor, common site being the anterior maxilla. Its origin from a dentigerous cyst and in the mandible is rare. A case of an AOT arising from a dentigerous cyst associated with an impacted permanent mandibular left lateral incisor is reported.     

Mandibular ameloblastoma. A review of the literature and presentation of six cases

Ameloblastoma is a benign odontogenic tumour of epithelial origin without induction in the connective tissue. In treating this type of tumour, it is important to assess the clinical type (solid, multicystic, unicystic, peripheral), localisation, and size of the tumour as well as age of the patient. Articles have recently been published with the purpose of providing updated knowledge and therapeutic approaches to ameloblastoma. We present six cases of patients with localised mandibular ameloblastoma who were treated during the last seven years. We present data on clinical appearance, histological characteristics, and therapeutic approach that was used, which included excision of the lesion, perilesional drilling of the bone, or block resection, according to the type of ameloblastoma. Lastly, we analysed follow-up measures and the rate of recurrence in these patients. The aim of this paper is to review the concepts relating to ameloblastoma that have been published recently and to assess their influence on the clinical attitude taken when facing this pathology, using our experience with ameloblastoma as a starting point to illustrate this discussion.     

Piezosurgical treatment of unicystic ameloblastoma

BACKGROUND: This case report describes a 22-year-old female who was referred with a radiolucent lesion between her mandibular right molars. The original radiographic image suggested an odontogenic cyst. METHODS: The surgical enucleation and osteotomy of the residual cavity were performed with a piezoelectric knife. Histologic analysis revealed the lesion was a unicystic ameloblastoma that showed no evidence of epithelial invasion in the connective tissue wall, and it appeared to be contained within the cystic cavity with an intraluminal projection showing features of an ameloblastoma. The patient refused any further surgical treatment. In consideration of the pathological result (no presence of invasive epithelial cells in the connective tissue wall of the cyst), a wait-and-see policy was proposed to the patient, with more extensive surgical treatment only if a recurrence was noted. RESULTS: The patient has been followed clinically and radiographically at 6-month intervals for 5 years. There have been no signs of recurrence. CONCLUSION: Therefore, the piezoelectric knife could be considered a promising device in the treatment of odontogenic tumors.