1/5. A classic adamantinoma arising from osteofibrous dysplasia-like adamantinoma in the lower leg: a case report and review of the literature. adamantinoma is known as a low-grade malignant biphasic tumor. Classic adamantinoma is in general characterized by admixture of both epithelial and osteofibrous components that are associated with various proportions and differentiation patterns. Osteofibrous dysplasia (OFD) is a self-limited benign fibro-osseous lesion of bone during infancy and childhood. OFD-like adamantinoma is characterized by predominance of osteofibrous tissues, in which small groups of epithelial cells are only detected by careful search or immunohistochemistry. There have been controversies as to the potential correlation among OFD, OFD-like adamantinoma and classic adamantinoma. We report an unusual case of adamantinoma arising in the tibia, with an extensive review of the literature. The present findings suggest a direct correlation between OFD-like adamantinoma and classic adamantinoma. At the age of 12 years, the tibial biopsy lesion was diagnosed as OFD. At the age of 23, the lesion became larger and more destructive on x-ray films. The biopsy lesion was diagnosed as classic adamantinoma. Wide excision was performed. The primary lesion was retrospectively diagnosed as OFD-like adamantinoma because of presence of keratin-positive epithelial cells within the stroma. At five years after surgery, the patient was free from recurrence or metastasis. The retrospective histological findings of OFD-like adamantinoma in the original biopsy and of a classic adamantinoma in all sections of the later resection specimen raised the possibility of an unusual progression of OFD-like adamantinoma to a classic adamantinoma. The present case and the literature review suggest that an OFD-like adamantinoma may be a precursor lesion of classic adamantinoma. Therefore, the possibility of progression of OFD-like adamantinoma to a classic adamantinoma should be kept in mind, particularly when the destructive changes are seen radiologically. ( info) |
2/5. Classical and atypical location of adamantinomas--presentation of two cases. BACKGROUND: Adamantinomas are rare low-grade primary bone tumors of unknown histological origin. They typically develop in the diaphyses and metaphyses of long bones, prevalently the tibia. case reports: The cases of 2 female patients, one with localized swelling of the lateral metatarsals and one with pain of the tibia are being reported. The diagnostic work-up including biopsy verified an adamantinoma in both cases. In the younger patient, the tumor was located in the metatarsals 4 and 5, in the older patient in the left tibial diaphyses. In both patients, an en-bloc resection of the tumor with wide surgical margins was performed. Further follow-ups 73 and 36 months after surgery showed no local recurrence or metastatic spread. CONCLUSION: Adamantinomas of the short bones are rarely reported. In accordance to the literature, an en-bloc resection with wide surgical margins seems to reduce the risk of local recurrence and metastatic spread. ( info) |
| OBJECTIVE: We report a patient with a cervicothoracic spinal and a mandibular adamantinoma. adamantinoma is a rare malignant neoplasm of bone and, to our knowledge, there have been only five cases of spinal adamantinoma reported. The pathogenesis of the adamantinoma, as well as the management of this extremely rare spinal tumor, is reviewed. CLINICAL PRESENTATION: A 55-year-old man was admitted to our service with cervical pain and signs of C8 and T1 radiculopathy. On physical examination, cervical spine deformity, swelling in the left mandible region, and signs of C8 and T1 radiculopathy were observed. Neuroradiology examinations showed an osteolytic mass of the C6, C7, and T1 vertebral bodies, extending into the lateral masses and transverse processes. After surgical procedures, the patient had clinical improvement. INTERVENTION: Corpectomy of C6, C7, and T1 was performed through a cervicothoracic anterior approach. Anterior stabilization of the spine was obtained using an autologous iliac crest graft and osteosynthesis with an anterior plate. On a second procedure, posterior tumor resection and spinal stabilization were performed. After the 1-year follow-up examination, a new anterior procedure was performed because of tumor recidivity and spine instability. CONCLUSION: adamantinoma, an extremely rare lesion, is a locally aggressive tumor with slow growth and the potential to metastasize. Although it is an extremely rare occurrence in the spine, adamantinoma should be considered on the diagnosis of tumors of the vertebrae. Neuroradiological examinations are not specific in the differentiation of this tumor from other conditions. This fact, coupled with the limited experience that most physicians in general have in dealing with this tumor, makes the diagnosis and treatment of adamantinoma challenging. ( info) |
4/5. Fibular osteoadiposal flap for treatment of tibial adamantinoma: a case report. We treated a case with left tibial adamantinoma by use of a contralateral fibular osteoadiposal flap. The donor site of conventional fibular osteocutaneous flap must be covered with a skin graft because if we close the donor skin defect directly, compartment syndrome might occur. We were able to close the donor skin defect because this combined type flap included only a small monitoring skin paddle. We present herein the utility of the osteoadiposal flap and show the value of a skin-sparing approach with a minimal aesthetic defect. ( info) |
5/5. Tibial adamantinoma with local recurrence and pulmonary metastasis: report of a case with histocytologic findings. BACKGROUND: adamantinoma is a rare primary bone neoplasm of low malignant potential that may recur or metastasize in a mall percentage of patients. The myriad histologic patterns may cause difficulty in distinguishing this tumor from other primary or metastatic neoplasms. The cytomorphologic findings of fine needle aspiration biopsy were reported previously in only a small number of cases. CASE: A 32-year-old man presented with a mass in the distal side of the left leg that was diagnosed as classic adamantinoma by open biopsy. Local recurrence and pulmonary metastases were confirmed by fine needle aspiration biopsy, which showed low grade, uniform cells with nuclear membrane grooves. The patient underwent a below-the-knee amputation and is receiving palliative treatment for progressive pulmonary spread. CONCLUSION: The diagnosis of adamantinoma requires knowledge of compatible clinical and radiologic studies as well as understanding of the variable histologic patterns that one may encounter. Fine needle aspiration biopsy is particularly useful in the diagnosis of recurrent and metastatic adamantinoma. This case report describes a distinctive cytomorphologic feature of nuclear grooves that may be a useful aid in distinguishing the tumor cells of adamantinoma from other cell types. ( info) |