Management of ameloblastic fibro-odontoma in a 6-year-old girl preserving the associated impacted permanent tooth

The ameloblastic fibro-odontoma (AFO) is a rare odontogenic tumor that occurs predominantly in children and is generally associated with unerupted teeth. The choice of treatment for this entity is conservative surgery with enucleation and its prognosis is excellent. However, preserving the associated impacted teeth may make complete removal of the lesion difficult and may explain some cases of recurrence. A case of AFO in a 6-year-old girl treated with enucleation and preservation of an impacted lower left first permanent molar is reported. After two years of follow-up, there were no signs of recurrence and complete spontaneous eruption of the preserved tooth was observed. The present report indicates that the degree of involvement of the impacted tooth by AFO is a fundamental aspect to be considered during radiological analysis and surgical assessment. We concluded that preservation of the impacted permanent teeth associated with this tumor should always be considered and carried out with caution, since they are clearly not included in the surgical cavity.     

Simultaneous adenomatoid odontogenic and keratocystic odontogenic tumours in a patient with Gorlin‐Goltz syndrome

Gorlin and Goltz described a syndrome in which multiple basal cell carcinomas, odontogenic keratocysts and bifid ribs occurred in combination. The jaw keratocysts are a consistent feature of ‘Gorlin‐Goltz’ or naevoid basal cell carcinoma syndrome. Central nervous system and ocular involvement occurred together with the fairly typical facial features of frontal bossing and hypertelorism. This case report documents the pathology associated with an impacted maxillary canine tooth in a boy with Gorlin‐Goltz syndrome. The patient presented for investigation of the failure of eruption of the right permanent maxillary canine tooth. Radiographic investigation showed the presence of a well circumscribed radiolucency located around the crown of an impacted right maxillary canine tooth. The patient's medical history revealed a medulloblastoma that was treated 13 years ago. The right maxillary canine tooth and associated peri‐coronal tissue were removed under general anaesthetic. A diagnosis of a keratocystic odontogenic tumour with an associated adenomatoid odontogenic tumour was made. The common differential diagnoses for a peri‐coronal radiolucency in the maxilla that need to be considered by dentists include a dentigerous cyst, follicular keratocystic odontogenic tumour and adenomatoid odontogenic tumour. A rare case of both keratocystic odontogenic tumour and associated follicular adenomatoid odontogenic tumour is described in a patient with naevoid basal cell carcinoma syndrome.     

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.     

Granular cell odontogenic cyst: a unicystic ameloblastoma with late recurrence as follicular ameloblastoma

This report is a follow-up of the original case of granular cell odontogenic cyst described by Gold and Christ in 1970. The lesion, originally treated by enucleation in 1965, recurred 18 years later as a follicular ameloblastoma with prominent plexiform and acanthomatous histologic patterns. No histologic evidence of granular cells was noted in the recurrent lesion. The recurrent ameloblastoma was treated by marginal resection of the body of the mandible and immediate reconstruction with an iliac bone graft. The anatomic restoration was excellent, and there is no recurrence 5 years postoperatively. This case supports the view that the granular cell odontogenic cyst is a unicystic (monocystic) granular cell ameloblastoma, and that the presence of granular cells in ameloblastoma is not a permanent feature and may be of little value as a prognostic indicator of aggressiveness.     

Dentigerous cyst containing multiple impacted teeth: a rare case report

The dentigerous cyst is a developmental odontogenic cyst, which apparently develops by accumulation of fluid between reduced enamel epithelium and the tooth crown of an unerupted tooth. It is one of the most prevalent types of odontogenic cysts associated with erupted, developing, or impacted tooth, particularly the mandibular third molars, the other teeth that are commonly affected are maxillary canines. The present case report is unique as it enclosed three permanent maxillary teeth: the central incisor, lateral incisor, and canine that had to be extracted during surgical enucleation of the cyst.     

Synchronous gubernacular canals with compound odontoma associated with a calcifying odontogenic cyst and transmigrated canine: An extremely rare event

This article reports the novel incidental concurrences of multiple oral lesions in a 13-year-old boy. Radiographically, an over-retained primary canine was seen with a subjacent mixed radiopacity and transmigrated permanent canine with a suspected dentigerous cyst (each with gubernacular canals). Treatment consisted of enucleation and removal of the impacted canine. The histopathologic findings were compound odontoma containing multiple tooth-like structures and calcifying odontogenic cyst with epithelium containing ghost cells, calcifications and subjacent fibrous connective tissue wall. All cases of odontomas should undergo microscopic assessment for diagnosis and ascertainment of associated pathosis.     

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.     

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.     

Adenomatoid Odontogenic Tumour Mimicking a Periapical Cyst in Pregnant Woman

Adenomatoid odontogenic tumours (AOT) are uncommon odontogenic lesions characterized histologi-cally by duct-like structures derived from the epithelial component of the lesion and can be distinctly classified into follicular, extrafollicular and extraosseous variants (Neville BW, Damm DD, Allen CM, et al. Adenomatoid Odontogenic Tumor. A Text Book for Oral and Maxillofacial Pathology, 2^nd edition, 621-3). Most of these tumours develop in the second or third decade of life and have a distinct predilection for women. The follicular variant accounts for 75% of reported cases (Curran AE, Miller EJ, Murrah VA. Adenomatoid odontogenic tumor presenting as periapical disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:557-60) and is associated with the crown of an impacted tooth, commonly the maxillary canine. We present a rare case of extrafollicular AOT mimicking a periapical cyst that originated in a woman in her first trimester of pregnancy and enlarged rapidly thereafter. The lesion was enucleated and sent for histopathology and immunohistochemistry, which revealed AOT with a cystic component with no dependence on oestrogen or progestrone for its growth. This case of AOT introduces us to the unique variation in its presentation and the difficulty in differentiation from periapical disease of inflammatory origin.     

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.     

Calcifying odontogenic cyst associated with complex odontoma: case report and review of the literature

We report a calcifying odontogenic cyst associated with odontoma (COCaO) and an included permanent canine in the superior maxilla, in a 19 year-old-man. The calcifying odontogenic cyst (COC) was first described as a distinct entity by Gorlin et al in 1962. The lesion is a mixed odontogenic benign tumor, and although most of the cases present cystic characteristics, a few are of the solid type (15%), and its rare malignant transformation is well documented. The COC may occur in association with other odontogenic tumors, the most common is the odontoma, occurring in about 24% of the cases. For this association the term Odontocalcifying odontogenic cyst has been suggested. Radiographically is a well defined mixed lesion and histologically consists of a large cyst. In the central area of the cyst enamel and dentin deposits can be found, irregularly distributed in areas and in other parts it takes on a well defined organoid aspect. A thorough review of literature takes place and the pathogenesis is discussed.     

Mandibular reconstruction using fresh frozen bone allograft after conservative enucleation of a mandibular odontogenic myxoma

The purpose of this article was to report the clinical, radiographic, and histological findings about a case of a young woman affected by a mandibular odontogenic myxoma. Conservative tumor resection was followed by immediate reconstructive treatment using fresh-frozen human bone graft, instead of autologous bone graft, as material for bone regeneration. Odontogenic myxoma, according to the World Health Organization, is classified as a benign tumor of mesenchymal origin whether or not containing odontogenic epithelium. Radiological and histological examination of the lesion confirmed the presence of an odontogenic myxoma, which was 21.2 mm high and 47.6 mm long; the lesion underwent biopsy evaluation before enucleation. According to literature and with the aim of a patient free of disease, conservative enucleation of the lesion was performed. The residual bone defect was filled with fresh-frozen bone allograft. At 6 months after surgery, no evidence of major complications was observed; the computed tomography scan revealed effective bone regeneration through the grafted area. The use of fresh-frozen bone allograft, thanks to its osteoinductive and osteoconductive properties, may represent an optional choice for reconstruction of bone defects after jaw tumor removal.     

Conservative management of a large keratocystic odontogenic tumor in the maxilla

Keratocystic odontogenic tumor is characterized by high recurrence rates. Conservative or aggressive management has been suggested as a method of treatment. Decompression is a conservative treatment that has been used in the treatment of large odontogenic cysts. The authors report a case of a 14-year-old patient with a keratocystic odontogenic tumor located in the right maxilla, which was treated by decompression followed by enucleation with curettage. The lesion did not recur on follow-up for 3 years after the enucleation surgery.     

Recurrent calcifying odontogenic cyst involving the maxillary sinus

The calcifying odontogenic cyst is an uncommon lesion that occurs in both jaws, however involvement of the maxillary sinus is rare. The accepted mode of treatment is enucleation with curettage since it is generally believed that recurrence following such treatment is extremely rare. Of the reported cases of recurrent calcifying odontogenic cysts, none have involved the maxillary sinus. This report is of a large recurrent calcifying odontogenic cyst involving the maxillary sinus, eroding the orbital floor as well as anterior and medial walls of the maxillary sinus and displacing an impacted upper canine into the nasal cavity, in a 45-year-old male patient, 8 years after the initial enucleation.     

Odontogenic myxoma: an orthodontic presentation

A maxillary odontogenic myxoma in a 17-year-old male was diagnosed as a result of his presenting to an orthodontist. Histological diagnosis was obtained following enucleation, and a further wide local excision performed as definitive treatment. The origin of the tumour was most likely from the follicular remnants of a previously exposed canine tooth.     

Hybrid odontogenic ghost cell tumor and cutaneous pilomatrixoma: a highly unusual coexistence

This article describes the first published case of coexistence in a child of a rare hybrid odontogenic ghost cell tumor and a solitary cutaneous pilomatrixoma. An 11-year-old boy presented with a large well-defined unilocular radiolucent lesion in the right posterior mandible. Marsupialization followed by enucleation of the remaining lesion at a later period was the treatment of choice. Histopathologic analysis revealed a hybrid tumor demonstrating areas identical to calcifying cystic odontogenic tumor, ameloblastoma, ameloblastic fibro-odontoma, ameloblastic fibromyxoma, and adenoid odontogenic tumor. A cutaneous nodule was also removed from the facial area and demonstrated classic features of pilomatrixoma on histopathology. Sixteen cases of hybrid calcifying cystic odontogenic tumor associated with odontogenic tumors other than ameloblastomas and odontomas are referred in the literature to date. Young males are frequently affected, and the mandible is the most common site of involvement. The occurrence in the same patient of 2 distinctive entities, which both demonstrate ghost/shadow cells, may be a coincidental finding or suggest a common origin regarding the histogenesis of these cells. Alternatively, future molecular studies may clarify possible genetic or/and predisposing factors for the development of these lesions.     

Trauma-induced dentigerous cyst involving the anterior maxilla

The dentigerous cyst is the most common developmental odontogenic cyst of the jaws. The purpose of this article was to report the case of a dentigerous cyst associated with a permanent maxillary central incisor in a 5-year-old boy who had suffered severe facial trauma at age 4. Panoramic and periapical radiographs revealed a well-defined radiolucent area with radiopaque borders surrounding the permanent maxillary left central incisor, causing displacement of the tooth buds of the permanent maxillary left lateral incisor and canine and root resorption of the deciduous maxillary left lateral incisor. Treatment was performed by aspiration and enucleation; the permanent maxillary left central incisor was part of the cyst and, thus, was also removed.     

Conservative approach to the treatment of keratocystic odontogenic tumor

The odontogenic keratocyst, also known as the keratocystic odontogenic tumor, is an aggressive, intraosseous lesion of odontogenic origin that presents a high rate of recurrence. Treatment modalities include aggressive surgical procedures and more conservative approaches that significantly influence the lesion's recurrence potential. The purpose of this case report was to demonstrate a conservative approach in the treatment of an extensive keratocystic odontogenic tumor, located in the mandible's posterior region, using decompression and enucleation.     

Adenomatoid odontogenic tumor: clinicopathologic and ultrastructural concepts

Two cases of adenomatoid odontogenic tumor have been presented, along with a review of the literature. One tumor had a rather typical presentation, occurring in a 12-year-old in association with an impacted canine tooth. Subsequent electron microscopic evaluation of this case provided corroboration of other recently published findings. In this case there was a striking accumulation of what could have been secretory granules at the pole of the cell nearest a "duct-like" space. This cell had ultrastructural characteristics in common with a preameloblast. The second case occurred in a more unusual situation, the patient being a 32-year-old man, with the AOT developing in the mandible between the roots of the canine and first premolar. Both patients were treated successfully by simple enucleation of the lesion.     

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.