REGISTRO DOI: 10.69849/revistaft/ch10202507271748
Philippi Machado dos Reis MSca,c*, Ana Clara Franco de Lima Espc, Diego de Carvalho Coelho Espc, Pedro Henrique Batista Santiago Ferreira Espc, Frederico Rodger Rodrigues Gomes MScc, Danuze Camila Peixoto Machado Espc, Rachel Santos das Chagas Espc, Ricardo Grillo PhDb,c
Abstract
Objectives: Ameloblastic fibrodontoma is a rare odontogenic lesion characterized by a combination of ectomesenchymal and epithelial odontogenic tissues, along with deposits of dentin and enamel. Its clinical and radiological presentation can often resemble other odontogenic lesions, complicating accurate diagnosis without supplementary methods like immunohistochemistry. This article aims to report a case of ameloblastic fibrodontoma, emphasizing the diagnostic challenges and the critical role of immunohistochemistry in ensuring accurate identification and appropriate clinical management.
A 23-year-old female patient with a history of thyroid cancer presented with a radiolucent lesion in the posterior mandible, discovered during a routine panoramic radiograph. A computed tomography scan was ordered, revealing a hypodense lesion (Fig. 1). Axial sections showed lingual cortical destruction (Figs. 2 and 3), while transverse sections (Fig. 4) demonstrated vestibular cortical fragility. The lesion was completely removed through osteotomy (Fig. 5 and 8). The surgical site was covered with collagen membranes (Fig. 7) and secured with a metal plate screwed into the vestibular aspect (Fig. 6). After an excisional biopsy, histological evaluation (Fig. 9) revealed mixed odontogenic epithelial structures and ectomesenchymal tissue, prompting immunohistochemical analysis. Immunohistochemical analysis showed positivity for cytokeratin and p40 (Fig. 10), confirming the diagnosis of ameloblastic fibrodontoma, due to the presence of enamel-like structures (Figs. 11 and 12). The patient is currently under regular follow-up and remains asymptomatic.
Discussion: Ameloblastic fibrodontoma presents clinical and radiological features that may overlap with other lesions, leading to potential underdiagnosis. The patient’s history of thyroid cancer may influence the lesion’s behavior, highlighting the need for thorough evaluation and further research. Excisional biopsy plays a crucial role in accurate diagnosis, allowing for detailed histopathological analysis and reducing the likelihood of recurrence. Immunohistochemical markers significantly enhance diagnostic accuracy, differentiating this lesion from others with similar features.
Conclusion: The case of ameloblastic fibrodontoma underscores the complexities of diagnosing rare odontogenic lesions. Immunohistochemical markers are essential for effective management and diagnosis, particularly in cases with prior systemic conditions. Continuous clinical follow-up is vital to monitor for recurrence or progression. This case emphasizes the need for increased awareness and further investigation into ameloblastic fibrodontoma.
Keywords: Oral, Pathology, Immunohistochemistry; Rare Diseases; Brazil; Fibroma
Introduction
Ameloblastic fibrodontoma is a rare odontogenic lesion characterized by a combination of ectomesenchymal and epithelial odontogenic tissues, along with deposits of dentin and enamel. Its clinical and radiological presentation can easily be mistaken for other odontogenic lesions, making accurate diagnosis challenging without the aid of supplementary methods such as immunohistochemistry. The diagnostic difficulties of these tumors arise from their resemblance to other benign odontogenic lesions, such as fibromas, compound odontomas, and complex odontomas (1).
The use of immunohistochemical markers is crucial for distinguishing between different odontogenic lesions. Recent studies have shown that antibodies such as SOX, p40, calretinin, and cytokeratin can improve diagnostic accuracy, as evidenced in a case report of a maxillary odontoma associated with Noonan syndrome. Although immunohistochemistry has been widely used for the diagnosis of malignant neoplasms, its role in identifying rare odontogenic tumors, such as ameloblastic fibrodontoma, is still being explored (2). A study by Best et al. (2024), which analyzed the aggressive presentation of ameloblastic fibrodontoma, found that this neoplasm can exhibit invasive characteristics that may suggest malignancy. As such, excisional biopsy followed by immunohistochemical analysis is essential for an accurate differential diagnosis, preventing inappropriate therapeutic approaches (3). Another relevant factor is the relationship between ameloblastic fibrodontoma and preexisting systemic conditions, such as a history of cancer, which can complicate the patient’s clinical presentation. Studies suggest that the presence of previous neoplasms may influence the biological behavior of odontogenic lesions, further complicating the diagnostic process (4).
The rarity of ameloblastic fibrodontoma also raises concerns about the standardization of clinical management due to the limited number of documented cases in the literature. The distinction between ameloblastic fibrodentinoma and ameloblastic fibrodontoma is frequently debated in the literature, with significant implications for treatment decisions (5). The aim of this article is to report a case of ameloblastic fibrodontoma, highlighting the diagnostic challenges and the critical role of immunohistochemistry in ensuring accurate identification and appropriate clinical management.
Case report
A 23-year-old female patient, with a history of thyroid cancer, underwent a total thyroidectomy five years ago. The patient has been on levothyroxine hormone replacement therapy since. During a routine radiographic exam, the general practitioner found a radiolucent lesion in the posterior region of the right mandible. The lesion appeared to be a cystic lesion. A computed tomography scan was ordered, revealing a hypodense lesion (Fig. 1). Axial sections showed lingual cortical destruction (Figs. 2 and 3), while transverse sections (Fig. 4) demonstrated vestibular cortical fragility. The patient was asymptomatic, with no pain or swelling.
Fig 3: Sequence of oblique transverse sections indicating the proximity of the tooth, the lesion’s closeness to the mandibular canal, and involvement of the cortical wall on the lingual side.
An excisional biopsy was performed under general anesthesia. The lesion was completely removed through osteotomy (Fig. 5 and 8). The surgical site was covered with collagen membranes (Fig. 7) and secured with a metal plate screwed into the vestibular aspect (Fig. 6). During the procedure, the impacted tooth associated with the lesion was also extracted. The patient was discharged the following day, but reported paresthesia in the right lower alveolar region. Initially, the lesion was thought to be an odontogenic fibroma due to its fibrous clinical features. However, histopathological analysis (Fig 9) revealed mixed odontogenic epithelial structures and ectomesenchymal tissue, with areas of dentin and enamel formation. This raised suspicion of a more complex lesion.
Fig 4: Immediate visualization after the complete removal of the lesion and tooth 48.
Fig 5: Immediate visualization after the installation of reinforcement for the cortical surface weakened by the lesion.
Fig 6: Supplementation of collagen membranes for covering areas with cortical involvement and as an addition to the granulated xenograft.
Fig 7: Total lesion with a fibronodular appearance measuring 6x5x6 mm at its greatest diameters.
Fig 8: Histological section stained with hematoxylin and eosin (H&E).
Immunohistochemistry was requested for a more precise diagnosis. The analysis showed positivity for cytokeratin and p40, markers associated with ameloblastic fibrodontoma (Figs 10, 11 and 12). Calretinin and SOX markers helped rule out other odontogenic lesions. Based on these findings, the final diagnosis was ameloblastic fibrodontoma.
Fig 9: Histological section resulting positive when subjected to monoclonal antibody p40 invasion.
Fig 10: Histological section indicating the presence of mineralized tissue associated with connective and epithelial tissue.
Fig 11: Histological section indicating the presence of tissue similar to dental enamel.
The patient is now under regular follow-up to monitor for recurrence and remains symptom-free. This case emphasizes the importance of immunohistochemistry in diagnosing rare odontogenic lesions and guiding appropriate treatment.
Discussion
Ameloblastic fibrodontoma is a rare mixed lesion composed of both odontogenic epithelial and mesenchymal tissue. Literature on this topic is scarce. This condition may present with characteristics similar to those of a complex odontoma, often leading to underdiagnosis due to clinical and radiological overlap. The rarity of this condition is highlighted by a study that reports only two cases of peripheral ameloblastic fibrodontoma. This underscores the need for further research to understand this neoplasm. The present case contributes to differential diagnosis through immunohistochemistry (5,6).
The patient’s history of thyroid cancer may have influenced the lesion’s behavior. Studies suggest a possible relationship between systemic diseases and the progression of odontogenic tumors. Previous neoplasms may alter the local tissue response. However, this association requires further investigation. Thyroidectomy and subsequent use of thyroid hormones may impact the odontogenic tissue response, but more research is needed to confirm this (4,7).
The importance of excisional biopsy is well documented. Surgical excision of complex odontogenic lesions allows for detailed histopathological analysis, which is crucial for differential diagnosis. Complete resection reduces the likelihood of recurrence. Continuous follow-up is essential due to the rarity of the lesion and potential for recurrence in some cases. In the present case, excisional biopsy provided a comprehensive evaluation of the tissue architecture, aiding in accurate diagnosis. (8,9).
The use of immunohistochemical markers, such as cytokeratin and p40, was crucial for differential diagnosis in this case. These markers allowed for the exclusion of other lesions with similar histological features. Immunohistochemistry is essential for the precise characterization of mixed odontogenic lesions. Advances in immunohistochemistry have significantly improved diagnostic accuracy for ameloblastic fibrodontoma, helping to distinguish this condition from other odontogenic lesions (6,8,10–12). Once diagnosed, this lesion requires prompt treatment to prevent malignant transformation (13).
Continuous clinical follow-up is critical due to the possibility of recurrence. Sanjai et al. (2022) highlight the progression of ameloblastic fibrodontoma to more aggressive forms. Although the patient has not shown signs of recurrence to date, periodic monitoring is essential for early detection of changes. Long-term follow-up is equally important. Ameloblastic fibrodontoma, although rare, can present aggressively, necessitating constant vigilance to monitor for potential recurrences or malignant transformations(14).
Conclusion
The presentation of ameloblastic fibrodontoma illustrates the intricate challenges involved in diagnosing rare odontogenic lesions. The use of immunohistochemical markers is crucial for distinguishing this condition from similar lesions, facilitating effective management. The patient’s history of thyroid cancer adds a layer of complexity, suggesting potential influences on the lesion’s behavior and highlighting the need for thorough clinical evaluations. Ongoing follow-up is essential to monitor for recurrence or aggressive progression. This case reinforces the importance of increased awareness and further research into ameloblastic fibrodontoma and its links to systemic conditions.
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aDepartment of Oral & Maxillofacial Surgery, Centro Universitário Estácio de Brasília, Brasília, Brazil.
bOral and Maxillofacial Surgery Training Program, Foundation of Dentistry – Fundecto, University of São Paulo, São Paulo-SP, Brazil.
c Departament of Oral & Maxilofacial Surgery, Faculdade Planalto Central, Brasília, Brazil.
* Corresponding author: Philippi Machados dos Reis, Orcid: 0009-0009-3817-8908, Centro Universitário Estácio de Brasília, e-mail: 1688.machado@gmail.com