Islands Of Odontogenic Epithelium Research Articles

Granular cell ameloblastoma: A rare and unique variant

Ameloblastoma is a slow-growing and locally invasive epithelial odontogenic tumor of the jaw that runs a benign course in most cases. Granular cell ameloblastoma is a rare subtype of ameloblastoma, showing the granular transformation of its cytoplasm. It is considered as an aggressive variant of ameloblastoma. Herein, we report the case of a 34-year-old male patient who presented with a swelling in the left cheek for a 1-year duration with a recent rapid increase in size. Examination showed a 4 × 3 cm growth involving the left buccal mucosa and retromolar area. Imaging studies showed expansile lytic lesion mandible. Biopsy revealed neoplasm with odontogenic epithelial islands showing peripheral palisading of tall columnar cells with reversal of polarity and the center of the islands showing stellate reticulum-like cells which were markedly replaced by granular cells. Granular cells can appear in various odontogenic and non- odontogenic tumors. When there is extensive granular cell change in ameloblastoma, it should be differentiated from other oral lesions with granular cells including granular cell odontogenic tumor, granular cell tumor, and congenital epulis.Herein we report the case of a 34-year-old male patient presented with swelling left cheek of one-year duration with recent rapid increase in size. Examination showed a 4x3 cm growth involving left buccal mucosa and retromolar area. Imaging studies showed expansile lytic lesion mandible. Biopsy revealed neoplasm with odontogenic epithelial islands showing peripheral palisading of tall columnar cells with reversal of polarity and the center of the islands showing stellate reticulum like cells which were markedly replaced by granular cells. Granular cells can appear in various odontogenic and nonodontogenic tumors. When there is extensive granular cell change in ameloblastoma it should be differentiated from other oral lesions with granular cells including granular cell odontogenic tumour, granular cell tumour and congenital epulis.

PERIPHERAL DENTINOGENIC GHOST CELL TUMOR: CASE REPORT

Peripheral dentinogenic ghost cell tumor (DGCT) is a rare benign mixed epithelial and mesenchymal odontogenic neoplasm, with approximately 52 cases reported in the literature. A 48-year-old woman was referred for evaluation of a painless, slow-growing swelling in the mandible caused by a removable denture that evolved over 1 year. Her medical history was unremarkable. Intraoral examination showed a pinkish, firm nodule, with erythematous areas, in the left mandibular alveolar ridge. Panoramic radiographic examination revealed no signs of bone involvement. Under the diagnostic hypothesis of inflammatory fibrous hyperplasia, an incisional biopsy was performed. Histopathologic analysis disclosed sheets and islands of odontogenic epithelium, with areas closely resembling ameloblastoma. Ghost cells, dentinoid material, and inflammatory foreign body reaction with multinucleated giant cells were also observed. The final diagnosis was peripheral DGCT. Treatment consisted of surgical excision, and no signs of recurrence were detected after 2 months of clinical follow-up.

PERIPHERAL AMELOBLASTOMA IN THE MAXILLARY GINGIVA: A RARE CASE

A 51-year-old man was referred to the Oral Medicine Clinic with an asymptomatic slow-growing palatal injury with a 15-year evolution history. Extraoral examination showed no gross abnormalities. Intraoral examination revealed a firm sessile nodule of normal coloration in the palatal mucosa adjacent to teeth #13 and #14, measuring approximately 2 cm. Imaging exams did not show bone changes. As diagnostic hypotheses, peripheral ossifying fibroma and inflammatory fibrous hyperplasia were considered. Excisional biopsy of the lesion was performed, and microscopic sections stained with hematoxylin and eosin revealed odontogenic epithelial islands in the oral lamina propria, composed of hyperchromatic columnar cells showing peripheral palisading, reverse polarization, and central stellate reticulum-like changes immersed in a dense connective tissue. Following clinicopathologic correlation, the diagnosis of peripheral ameloblastoma was established. Follow-up is ongoing and no recurrence has been detected so far.

HISTOLOGIC AND RADIOGRAPHIC STUDY OF PATHOLOGIC CHANGE IN COMPLETE IMPACTED THIRD MOLARS DENTAL FOLLICLES

Background: Prophylactic extraction of the asymptomatic impacted third molar is routinely practiced in Europe and the United States. The justification for prophylactic extraction includes the need to reduce the risk of pathologic changes such as cysts and tumors. 
 Objectives: This study aimed to study the histological and radiological changes in the tooth follicles of upper and lower complete impacted 3rd molars -which appeared radiologically normal.
 Material and method: A prospective study included fifty patients aged 20 years and above who were referred to the Oral Surgery Clinic, Faculty of Dentistry, University of Sana'a. Patients had follicular space between (2.5mm -3mm) as measured by the panoramic X-ray. These teeth were removed surgically and the follicle was sent for histopathological examination.
 Results: Most histopathological changes were in dental follicles with a size of <2.5 mm (86%), and only 14% with 2.5 mm - 3 mm. There was statistical significance between the smallest size of dental follicles with the incidence of pathological histological changes indicating a high probability of developing neoplasm (p =0.008). Of the 50 follicular patients, 28% showed HC, nine (64%) had ameloblastoma, four (29%) had a dentigerous cyst, and only one case (7%) had a multicalcified focus with islands of odontogenic epithelium. While 72% of the samples had normal follicles and non-specific chronic inflammatory cells. There is an association between female sex and pathological histological changes (12 females: 2 males, p =0.008), age group 21-25 years (93% HC), with mandibles (65% HC). Regarding angle and histopathological changes, 36% were vertical, 29% mesioangular, 14.2% horizontal and destioangular, and 7.1% buccoangular.
 Conclusion: High incidence of HC occurred in patients with DF, and it was associated with smaller dental follicle size, most HC was ameloblastoma, followed by dentigerous cyst, while 72% of samples had normal follicles and non-specific chronic inflammatory cells. There is a correlation between female gender, younger age group, and jaw position with HC. Prophylactic extraction of the asymptomatic impacted third molar should be routinely practiced in Yemen, to reduce the risk of pathological changes, especially in females and younger age groups.
 
 Peer Review History: 
 Received 11 January 2021; Revised 8 February; Accepted 28 February, Available online 15 March 2021
 UJPR follows the most transparent and toughest ‘Advanced OPEN peer review’ system. The identity of the authors and, reviewers will be known to each other. This transparent process will help to eradicate any possible malicious/purposeful interference by any person (publishing staff, reviewer, editor, author, etc) during peer review. As a result of this unique system, all reviewers will get their due recognition and respect, once their names are published in the papers. We expect that, by publishing peer review reports with published papers, will be helpful to many authors for drafting their article according to the specifications. Auhors will remove any error of their article and they will improve their article(s) according to the previous reports displayed with published article(s). The main purpose of it is ‘to improve the quality of a candidate manuscript’. Our reviewers check the ‘strength and weakness of a manuscript honestly’. There will increase in the perfection, and transparency. 
 Received file: Reviewer's Comments:
 Average Peer review marks at initial stage: 5.5/10
 Average Peer review marks at publication stage: 7.5/10
 Reviewer(s) detail:
 Dr. A.A. Mgbahurike, University of Port Harcourt, Nigeria, amaka_mgbahurike@yahoo.com
 Dr. Alfonso Alexander Aguileral, University of Veracruz, Mexico, aalexander_2000@yahoo.com
 
 Similar Articles:
 RADIOGRAPHIC ASSESSMENT OF THE COURSE AND VISIBILITY OF THE MANDIBULAR CANAL BY PANORAMIC RADIOGRAPHY

REGIONAL ODONTODYSPLASIA AFFECTING THE MAXILLA: A CASE REPORT

Regional odontodysplasia (RO) is a rare dental anomaly of unknown etiology that can affect both deciduous and permanent dentition. We report a case of a 10-year-old female child with a chief complaint of painless swelling in the right maxilla, extending from the region of the canine to the tuberosity, covered by normal-colored mucosa, and approximately 2.5 cm in diameter. A panoramic radiograph showed malformation of the mineralized tissues of teeth of this region, with quite broad pulp chambers, suggesting the appearance of ghost teeth. An incisional biopsy of the soft tissue lesion was performed. Microscopic examination revealed the presence of small calcified enameloid conglomerates associated with spindle cells arranged in a storiform pattern, small islands of odontogenic epithelium, and hyalinized dentin-like material. The diagnosis was RO. The soft tissue lesion was surgically removed, and the patient was referred to a dentistry service for further functional rehabilitation of the affected teeth.

CENTRAL ODONTOGENIC FIBROMA IN THE MANDIBLE: A CASE REPORT

Central odontogenic fibroma (COF) is a rare, benign mesenchymal odontogenic neoplasm that has a wide patient age range and a relatively similar distribution in the maxilla and mandible. A 60-year-old woman was referred for evaluation of a painless, slow-growing swelling in the mandible, extending from the left ramus to the distal root of tooth #36, covered by normal mucosa. Computed tomography showed an expansive, unilocular hypodense lesion with hyperdense foci, associated with unerupted tooth #38, measuring approximately 4.5 cm in diameter. Under the diagnostic hypothesis of calcifying epithelial odontogenic tumor, an incisional biopsy was performed. Histopathological analysis disclosed a moderately cellular, fibroblastic connective tissue intermixed with inactive-looking odontogenic epithelial islands and strands. The definitive diagnosis was COF. The treatment consisted of enucleation, curettage, and extraction of the associated tooth. No signs of recurrence were observed after 14 months of surgical excision. The patient remains under clinical and radiographic follow-up. Central odontogenic fibroma (COF) is a rare, benign mesenchymal odontogenic neoplasm that has a wide patient age range and a relatively similar distribution in the maxilla and mandible. A 60-year-old woman was referred for evaluation of a painless, slow-growing swelling in the mandible, extending from the left ramus to the distal root of tooth #36, covered by normal mucosa. Computed tomography showed an expansive, unilocular hypodense lesion with hyperdense foci, associated with unerupted tooth #38, measuring approximately 4.5 cm in diameter. Under the diagnostic hypothesis of calcifying epithelial odontogenic tumor, an incisional biopsy was performed. Histopathological analysis disclosed a moderately cellular, fibroblastic connective tissue intermixed with inactive-looking odontogenic epithelial islands and strands. The definitive diagnosis was COF. The treatment consisted of enucleation, curettage, and extraction of the associated tooth. No signs of recurrence were observed after 14 months of surgical excision. The patient remains under clinical and radiographic follow-up.

AMELOBLASTIC FIBRODENTINOMA INVOLVING THE POSTERIOR MAXILLA: A CASE REPORT

Ameloblastic fibrodentinoma (AFD) is a rare benign odontogenic tumor, which can mislead the clinician to a variety of differential diagnosis. A 5-year-old white man presented with a painless swelling of the left maxillary region of 1-year duration. A cone beam CT revealed a hypodense unilocular lesion with well-delimited borders involving the impacted upper molars and the left tuberosity region. Differential diagnoses of adenomatoid odontogenic tumor, calcifying epithelial odontogenic tumor, ameloblastoma, and ameloblastic fibroma were considered. After incisional biopsy, the histopathologic sections showed fragments of connective tissue containing islands and cords of odontogenic epithelium with peripheral cells resembling ameloblasts. The lesion was surgically removed, and the histopathologic examination showed deposition of dentinoid material in contact with the islands of odontogenic epithelium. A diagnosis of AFD was made. Currently, the patient is under observation and without recurrence.

AMELOBLASTOMA IN THE MAXILLA: CLINICAL, RADIOLOGIC, AND PATHOLOGIC ANALYSIS

Ameloblastoma is the most common odontogenic tumor; however, its occurrence in the maxilla is rare. Locally it shows infiltrative behavior and it has high rates of relapse. A 43-year-old male presented with an oral sinus communication after extraction of tooth #26. Oral examination revealed a 4-cm nodular lesion in the posterior region of the left superior vestibular sulcus, with an irregular surface and soft consistency. Computed tomography showed complete opacification as well as the destruction of the floor and the posterior wall of the left maxillary sinus. An incisional biopsy was performed and histopathologic examination revealed cords and islands of odontogenic epithelium embedded in a stroma with dense connective tissue. Basal cells showed cytoplasmic vacuolization and reverse polarization of the nuclei. The cells in the central region were loosely arranged and there were foci of cystic degeneration. After diagnosis of ameloblastoma, the lesion was enucleated and no recurrence was observed during follow-up.

EXTENSIVE AMELOBLASTIC FIBROMA IN A 4-YEAR-OLD GIRL: A CASE REPORT

Ameloblastic fibroma (AF) is an uncommon mixed odontogenic tumor that usually occurs in the first 2 decades of life with slightly higher prediction for boys and men. We report a case of AF in a 4-year-old girl who presented with a chief complaint of swelling in the right mandibular posterior region. The computed tomography showed a large multilocular hypodense area extending from mandibular ramus to parasymphysis on the same side, causing dental retention. A presumptive preoperative diagnosis of ameloblastoma was made. An incisional biopsy was performed, and anatomopathologic analysis showed odontogenic epithelial islands and cords in a primitive connective tissue stroma resembling the dental papilla. These findings confirmed the diagnosis of AF. The patient was referred for enucleation and remains in clinical follow-up.

REGIONAL ODONTODYSPLASIA: A CASE REPORT

Objective Report a case of regional odontodysplasia in the maxilla of a pediatric patient and the immunohistochemical expression of different types of collagen. Regional odontodysplasia is a rare developmental anomaly, involving the ectoderm and ectomesenchyme of the temporally and permanent teeth. It tends to be locallizated in only one arch of the jaws. Case report A 8-year-old female patient, with no significant hereditary or pathological history. On examination dental agenesis C, D, E is observed. Radiographically revealed 6, 7 and 8 with radiopaque contour and loss of the delimitation between enamel-dentin complex, giving an appearance of ghost teeth. Surgical treatment was performed. Microscopic examination follicular tissue contains scattered collection of enameled congregates and islands of odontogenic epithelium. Immunohistochemical expression of different types of collagens was heterogeneous in the dentin (col. 1,2,3,4,5,6,10 and 11). These results are consistent with an abnormal dentin development. Conclusion Regional odontodysplasia is an alteration that develops at a very early age, so the interdisciplinary management and the choice of treatment are fundamental since the treatment must be specific for each patient.

Yes-associated protein (YAP) molecule expression in odontogenic epithelium of ameloblastoma

YAP (Yes-associated protein molecule) augments multiple processes known to be important for tumor progression and metastasis. Hence, the application of YAP as a targeted therapy for several malignancies has been documented in the literature. Ameloblastoma being a locally invasive odontogenic lesion causes tissue destruction and presents with a great chance of recurrence. Based on these qualities, we hypothesized that YAP could exhibit increased expression in the odontogenic epithelium of ameloblastoma. Therefore, the objective of the present study was to evaluate the immunohistochemical expression of YAP in ameloblastoma and compare the immunoreactivity of YAP in ameloblastoma and dental follicle. Twenty-four paraffin-embedded tissue blocks of ameloblastoma and dental follicle each were retrieved from the departmental archives. Immunohistochemical (IHC) staining was performed employing YAP antibody on 5-μm thick sections. The expression of YAP was reviewed on the peripheral lining of ameloblast-like cells and centrally placed stellate reticulum-like cells and was further graded as negative, weak, or strong. For data analysis, Chi-square test and Fisher's exact test were applied. In ameloblastoma, peripheral/basal cells exhibited strong expression for YAP in 12/24 cases and weak expression in the other 12 cases. Whereas, 20 cases of dental follicle presented negative expression and only four cases exhibited weak expression (p < 0.001). On statistical analysis, the differences in expression of YAP in stellate cells of ameloblastoma and dental follicle were found to be significant (p < 0.001). YAP expression was strongly seen in ameloblastoma as compared to the epithelial islands of dental follicle. The neoplastic nature of ameloblastoma could be attributed to YAP molecule and it may play a possible role in tumor invasiveness. Positive YAP expression in ameloblastoma and negative expression in the odontogenic epithelial islands of dental follicle insinuate that YAP could be responsible for the aggressiveness and local tissue invasion commonly encountered with ameloblastoma.

Focal Adhesion Kinase Expression in Ameloblastoma: A Preliminary Observational Study.

Focal Adhesion Kinase (FAK) is a membrane-associated molecule, which plays a major role in tumourigenesis by promoting cell survival, proliferation, migration, and invasion. Hence, FAK is a well known molecule that has been targeted therapeutically for the treatment of various malignancies. Ameloblastoma (AB) is a locally invasive, destructive and recurrent benign odontogenic lesion. Based on these properties, we have hypothesized that odontogenic epithelium of AB could show increased expression of FAK. To study the expression of FAK in odontogenic epithelium of AB using immunohistochemistry. Thirty four paraffin embedded AB and 17 Dental Follicle (DF) tissues were retrieved from archives. Immunohistochemical staining was performed using FAK antibody on 5 μm thick formalin fixed paraffin embedded sections. The expression of FAK was studied in ameloblast like cells and stellate reticulum-like cells and was graded as negative, weak and strong. The data was analysed using chi-square test and Fisher's-exact test. In AB, both ameloblast like and stellate reticulum like cells showed positive expression of FAK in the cytoplasm in all the cases (34). The epithelial islands from all the DFs (17) were negative for FAK expression. The difference in the expression of FAK in ameloblast like and stellate reticulum like cells was statistically significant (p<0.0001). Statistically significant difference was observed in the expression of FAK in stellate reticulum like cells of AB and odontogenic epithelial islands of DF (p = 0.0017). All the AB cases showed strong expression of FAK in ameloblast like cells and negative expression was seen in all the cases of DF. FAK was strongly expressed in AB as compared to epithelial islands of DF. FAK could be responsible for the neoplastic nature of AB and possibly play a role in tumour invasiveness.

Ameloblastic fibro-odontoma: A diagnostic challenge

Ameloblastic fibro-odontoma (AFO) is a rare mixed odontogenic tumor. It occurs predominantly in children and young adults with no sex predilection, mostly in the mandible posterior region. A painless swelling is the most common clinical sign of AFO. Radiographically, AFO shows a circumscribed radiolucency, which contains radiopaque foci of various sizes and shapes. Histological examination reveals a fibrous soft tissue, islands of odontogenic epithelium, and a disordered mixture of dental tissues. Treatment of AFOs usually consists of enucleation or surgical curettage, which is possible due to their benign biological behavior. Here, we are reporting a case of AFO in a 20-year-old male.

Basal Cell Ameloblastoma: Report of a Rare Case with Review of Literature

Ameloblastoma is a benign tumour of odontogenic epithelial origin with several histological variants comprising of follicular, plexiform, acanthomatous, desmoplastic, granular cell and basal cell types. Basal cell ameloblastoma (BCA) is the rarest histologic subtype in which the tumour is composed of numerous odontogenic epithelial islands supported by fibrous connective tissue stroma. It exhibits variations in the clinical course and histological features compared with the classic type of ameloblastoma. Till date, only 11 cases of BCA have been reported in the literature. Considering the rarity of the lesion, we report here a unique case of BCA occurring in the 8th decade with review of the literature of this rare variant.

A Histopathological and Immunohistochemical Analysis of Ameloblastic Fibrodentinoma

Ameloblastic fibrodentinoma (AFD) is considered a mixed odontogenic tumor that is characterized by conserved epithelial and ectomesenchymal neoplastic components. AFD is composed of long narrow cords and islands of odontogenic epithelium; the epithelial strands lie in a myxoid cell-rich ectomesenchymal tissue with stellate-shaped fibroblasts that exhibit long slender cytoplasmic extensions that resemble dental papilla. The lesions show the presence of dysplastic dentin. Although AFD is a rare entity and its very existence is not completely accepted, based on the extent of histodifferentiation, it is considered to represent a stage between ameloblastic fibroma and ameloblastic fibroodontoma. This study aimed to provide a histopathological and immunohistochemical characterization of this infrequent tumor. A large panel of antibodies including amelogenin, Ck 19, calretinin, syndecan-1, E-cadherin, MSH2, histone H3, and Ki-67 was used to illustrate the nature of the tumor.

Odontogenic Fibroma, Including Amyloid and Ossifying Variants

Sixty-five cases of odontogenic fibroma (OdonF) are herein presented having been segregated into peripheral, extra bony tumors (n=40) and tumors arising in bone or centrally (n=25). All cases were characterized microscopically by a fibrous proliferation that varied within and between cases in cellularity and collagen fibril diameter, with intermixed odontogenic epithelial islands and cords. All central lesions presented as well demarcated radiolucencies and resorption of contiguous tooth roots was a common finding. These intraosseous lesions were of the WHO type; the so-called nonWHO type was excluded as all lesions with this diagnosis were devoid of an epithelial component and could be reclassified as other soft tissue fibrogenic tumors. Neither the central tumors nor the peripheral lesions recurred following enucleation/curettage, with a mean follow-up of 4 and 3.4years respectively. Three distinct microscopic variations were encountered in this series: (1) two cases of OdonF with giant cell reaction, (2) two instances of OdonF with ossifying fibroma; and (3) four instances of OdonF with odontogenic ameloblast-associated protein (ODAM), an amyloid-like protein found deposited adjacent to epithelial cords plus CD1a+/S-100+ Langerhans dendritic cells entwined around the epithelial element. A single instance of the odontogenic fibroma-like hamartoma/enamel hypoplasia syndrome has been included in this series.

Central odontogenic fibroma: new findings and report of a multicentric collaborative study

The aim of this study was to describe the clinicopathologic and immunohistochemical characteristics of 14 cases of central odontogenic fibroma (COF), and the ultrastructural features of 2 of them. Collaborative retrospective study based on the records of 4 oral pathology diagnostic services in Latin America based on the current World Health Organization classification. There were 7 male and 7 female patients (mean age 31.8 years). Eight tumors occurred in the maxilla and 6 in the mandible. Thirteen cases were epithelium-rich and 1 epithelium-poor COF. Three were classified as hybrid COF with giant cell lesion. Mean size of the hybrid lesions were larger than pure COF (3.8 vs. 2.4 cm). Odontogenic epithelial islands were immunoreactive for cytokeratin (CK) AE1/AE3, CK5, CK14, CK19, and 34BE12 and negative for CK1 and CK18. Langerhans cells positive for S-100 and CD1a were found within the epithelial islands in 6/6 tested cases. CD68 was expressed in the giant cells of the hybrid lesions and in a few mononuclear cells of 2 cases of COF. Ki-67 index was <1% in all cases. In 6 tumors (42.8%), there were small globular eosinophilic droplets within the epithelial islands, which were positive for collagen type IV, and 9/13 cases (69.2%) were focally positive for smooth muscle actin. In addition to fibroblasts, myofibroblastic differentiation was found in the 2 cases studied ultrastructurally. Immunohistochemistry was useful to confirm the presence of epithelium and to exclude other central fibrous tumors. COF also contains a variable number of mast cells, Langerhans cells, and myofibroblasts, and further studies are needed to better understand the participation of these cells in COF histogenesis.

Ameloblastic fibro-odontoma masquerading as odontoma

Ameloblastic fibro-odontoma (AFO) is a rare, benign epithelial mixed odontogenic tumor. It occurs as an intraosseous lesion, generally asymptomatic and more prevalent in children and adolescent. AFO is found on radiographic evaluation of patients with unerupted or impacted teeth in many cases. Histological examination reveals a fibrous soft tissue, islands of odontogenic epithelium and a disordered mixture of dental tissues. The treatment modality in most cases involves conservative surgery with enucleation. We present a case of 13-year-old boy with a missing right central incisor, mimicking-like odontoma on radiograph but proved to be AFO and treated with enucleation with preservation of impacted tooth. There was no recurrence after one year of follow-up. This report discusses the clinical, radiographical, histological features and surgical assessment to preserve the impacted tooth associated with AFO.

二期的に摘出術を施行し下顎形態を温存した歯原性粘液腫の1例

Odontogenic myxoma is a comparatively rare lesion of the jaw bone. A case of odontogenic myxomaoccurring in the mandible of a young girl that was treated by two-stage surgery to preserve the form of themandible is presented. A 12-year-old girl was referred to our hospital because of a painless swelling in the rightlower molar region. Radiographic examinations revealed multilocular radiolucency with a fine“soap bubble”appearance, accompanied by impacted second and third molars, at presentation. As much as possible of the tumor was removed by an intraoral approach with the patient under general anesthesia. To conserve jaw and mouth function, segmental resection was not performed. Histological examination revealed a tumor consisting of proliferative spindle cells without nuclear atypia in an abundant myxoid stroma. Islands of odontogenic epithelium were found within the tumor. A pathologic diagnosis of odontogenic myxoma was made. Three years later, secondary surgical removal of the tumor was carried out after the bone had regenerated. At present, 3 years after secondary surgery, bone has formed in the cavity remaining after the operation, and the tumor has not recurred. The patient will be carefully observed for tumor recurrence hereafter.

Maxillary ameloblastic fibroma: a case report

Ameloblastic fibroma is a relatively rare benign odontogenic tumor in which both the epithelial and ectomesenchymal components are neoplastic. An 8-year-old Caucasian boy was referred to the dentist for evaluation of failed eruption of the maxillary left first molar. The panoramic radiograph showed a well-circumscribed unilocular radiolucency involving an unerupted maxillary left first permanent molar. The lesion was enucleated and the material was sent for histopathologic examination. Microscopically, it was composed by cords and islands of odontogenic epithelium in a myxoid cell-rich stroma that closely resemble the dental papilla with histopathological diagnosis of ameloblastic fibroma. After 24 months of follow-up no recurrence was observed and the maxillary left first molar erupted spontaneously through the buccal mucosa and was aligned with a fixed orthodontic appliance. This case emphasized the importance of careful differential diagnosis of intraosseous oral lesions and reported a rarity of the lesion and its atypical location.