PubMed · 14830121
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C A R SCHULENBURG. 1951. Adamantinoma.. https://pubmed.ncbi.nlm.nih.gov/14830121/
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BACKGROUND: Adamantinoma is a rare tumor of long bones that can be mistaken for a carcinoma, and numerous other lesions such as myoepithelial tumor, malignant mixed tumor of bone, fibrous dysplasia/osteofibrous dysplasia, and vascular tumors. METHODS: The histology and clinical features of 12 cases accrued from the surgical pathology files over a period of 23 years were analyzed. RESULTS: Of the 12 cases, 4 were men and 8 women, with a mean age of 30 years. Radiologically the tumor presented as a lytic, eccentric, intracortical lesion in the tibia. The histology revealed epithelial islands with basaloid, spindle, and squamous features on a background of fibrotic stroma. Three patients came back with local recurrences, and one of them developed pulmonary metastasis. DISCUSSION: In this article, an attempt is made to study the morphology, biology, and the differential diagnoses of adamantinoma of long bones.
A 26-year-old female with a tibial lesion diagnosed as an adamantinoma was treated with intra-lesional curettage, bone grafting and intra-medullary nailing. Six years post-surgery, she presented with an asymptomatic primary site but with a metastatic lesion in the mid-shaft of the ipsilateral femur and lung metastases. The femoral lesion was treated with wide excision and reconstructed with an allograft and plate fixation. Pulmonary metastatectomy was carried out for the lung lesions. A follow-up CT scan of the chest at 1 year after the surgery for the metastatic lesions revealed fresh unresectable bilateral metastases. Although cases of local recurrences and pulmonary metastases in adamantinoma are reported, this case is unusual in presenting without a local recurrence but with simultaneous skeletal and pulmonary metastases.
The outcome of tibial allograft reconstruction after resection of a tumour is inconsistent and has a high rate of failure. There are few reports on the use of tibial allografts in children with open growth plates. We performed 21 allograft reconstructions (16 osteoarticular, five intercalary) in 19 consecutive patients between seven and 17 years of age. Two had Ewing's sarcoma, one an adamantinoma and 16 osteosarcoma, one with multifocal disease. Five patients have died; the other 14 were free from disease at the time of follow-up. Six surviving patients (eight allograft reconstructions) continue to have good or excellent function at a mean of 59 months (14 to 132). One patient has poor function at 31 months. The other seven patients have a good or excellent function after additional procedures including exchange of the allograft and resurfacing or revision to an endoprosthesis at a mean of 101 months (43 to 198). The additional operations were performed at a mean of 47 months (20 to 84) after the first reconstruction. With the use of allograft reconstruction in growing children, joints and growth plates may be preserved, at least partially. Although our results remain inconsistent, tibial allograft reconstruction in selected patients may restore complete and durable function of the limb.