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Metastatic ameloblastoma responding to combination chemotherapy: case report and review of the literature.

Amzerin M, Fadoukhair Z, Belbaraka R, Iraqui M, Boutayeb S, M'rabti H, Kebdani T, Hassouni K, Benjaafar N, El Gueddari BK, Errihani H - J Med Case Rep (2011)

Bottom Line: After two years he developed multiple lung metastases.Our patient received a combination chemotherapy using doxorubicin and cisplatin.Only through continuous reporting of such cases will clinicians be able to draw an optimal strategy for management of this pathology.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Medical Oncology, National Institute of Oncology, Rabat, Morocco. mounia.amzerin@gmail.com.

ABSTRACT

Background: Ameloblastoma is a rare benign odontogenic tumor with locally aggressive behavior and a high recurrence rate. When metastases occur, which are uncommon, lungs constitute the most frequent site involved. Malignant ameloblastomas are different from ameloblastic carcinomas. Malignant ameloblastomas are tumors considered metastatic despite the appearance of well-differentiated or benign histology, while ameloblastic carcinomas are histologically malignant in both primary and metastatic sites.

Case presentation: A 24-year-old Moroccan man presented a malignant ameloblastoma of the mandible. The tumor was entirely resected. Five years later, a local recurrence occurred. Our patient was treated by exclusive radiotherapy with persistence of a residual disease. After two years he developed multiple lung metastases. Our patient received a combination chemotherapy using doxorubicin and cisplatin.

Conclusion: Less than 50 cases of ameloblastoma with metastases have been reported. There is still no standard treatment for metastatic ameloblastoma. Only through continuous reporting of such cases will clinicians be able to draw an optimal strategy for management of this pathology.

No MeSH data available.


Related in: MedlinePlus

Histopathologic features of ameloblastoma.
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Figure 1: Histopathologic features of ameloblastoma.

Mentions: A 24-year-old Moroccan man presented in 2000 with a mass of the right mandible. A panoramic radiograph revealed a multilocular radiolucency, requiring a biopsy. A histopathological examination of the specimen showed a well-differentiated neoplastic proliferation. This appeared as strands of peripheral columnar cells in palisading orientation. The fibroblastic tumor-associated stroma was dense with collagen fibers and highly infiltrated by inflammatory mononuclear cells. No histological signs of malignancy were observed (Figure 1). The diagnosis of ameloblastoma was confirmed. Treatment consisted of hemimandibulectomy. Surgical margins were free of tumor. Five years later, the lesion recurred as a mass of his right jaw. The recurrence was confirmed by a second biopsy. At the same time, a chest tomography revealed three metastatic nodules of lungs. Our patient received exclusive radiotherapy for the jaw mass, at the dose of 60 Gy. No treatment was delivered for the lung metastases. The disease was controlled for two years, until our patient presented again with a right submandibular mass and multiple bilateral lung metastases (Figure 2A). Our patient received combination chemotherapy using doxorubicin 50 mg/m2 and cisplatin 100 mg/m2. The assessment of response to chemotherapy was made after two cycles. The pain disappeared, and tomography showed, according to RECIST criteria, local stabilization and partial response of the lung lesions (30%) (Figure 2B). The response was maintained after six cycles of chemotherapy.


Metastatic ameloblastoma responding to combination chemotherapy: case report and review of the literature.

Amzerin M, Fadoukhair Z, Belbaraka R, Iraqui M, Boutayeb S, M'rabti H, Kebdani T, Hassouni K, Benjaafar N, El Gueddari BK, Errihani H - J Med Case Rep (2011)

Histopathologic features of ameloblastoma.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3198715&req=5

Figure 1: Histopathologic features of ameloblastoma.
Mentions: A 24-year-old Moroccan man presented in 2000 with a mass of the right mandible. A panoramic radiograph revealed a multilocular radiolucency, requiring a biopsy. A histopathological examination of the specimen showed a well-differentiated neoplastic proliferation. This appeared as strands of peripheral columnar cells in palisading orientation. The fibroblastic tumor-associated stroma was dense with collagen fibers and highly infiltrated by inflammatory mononuclear cells. No histological signs of malignancy were observed (Figure 1). The diagnosis of ameloblastoma was confirmed. Treatment consisted of hemimandibulectomy. Surgical margins were free of tumor. Five years later, the lesion recurred as a mass of his right jaw. The recurrence was confirmed by a second biopsy. At the same time, a chest tomography revealed three metastatic nodules of lungs. Our patient received exclusive radiotherapy for the jaw mass, at the dose of 60 Gy. No treatment was delivered for the lung metastases. The disease was controlled for two years, until our patient presented again with a right submandibular mass and multiple bilateral lung metastases (Figure 2A). Our patient received combination chemotherapy using doxorubicin 50 mg/m2 and cisplatin 100 mg/m2. The assessment of response to chemotherapy was made after two cycles. The pain disappeared, and tomography showed, according to RECIST criteria, local stabilization and partial response of the lung lesions (30%) (Figure 2B). The response was maintained after six cycles of chemotherapy.

Bottom Line: After two years he developed multiple lung metastases.Our patient received a combination chemotherapy using doxorubicin and cisplatin.Only through continuous reporting of such cases will clinicians be able to draw an optimal strategy for management of this pathology.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Medical Oncology, National Institute of Oncology, Rabat, Morocco. mounia.amzerin@gmail.com.

ABSTRACT

Background: Ameloblastoma is a rare benign odontogenic tumor with locally aggressive behavior and a high recurrence rate. When metastases occur, which are uncommon, lungs constitute the most frequent site involved. Malignant ameloblastomas are different from ameloblastic carcinomas. Malignant ameloblastomas are tumors considered metastatic despite the appearance of well-differentiated or benign histology, while ameloblastic carcinomas are histologically malignant in both primary and metastatic sites.

Case presentation: A 24-year-old Moroccan man presented a malignant ameloblastoma of the mandible. The tumor was entirely resected. Five years later, a local recurrence occurred. Our patient was treated by exclusive radiotherapy with persistence of a residual disease. After two years he developed multiple lung metastases. Our patient received a combination chemotherapy using doxorubicin and cisplatin.

Conclusion: Less than 50 cases of ameloblastoma with metastases have been reported. There is still no standard treatment for metastatic ameloblastoma. Only through continuous reporting of such cases will clinicians be able to draw an optimal strategy for management of this pathology.

No MeSH data available.


Related in: MedlinePlus