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      Treatment of Osteofibrous Dysplasia and Associated Lesions

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          Abstract

          Purpose

          To report long term treatment outcomes of osteofibrous dysplasia and association with adamantinoma.

          Patients and Methods

          From January 1984 to July 2001, 14 patients with osteofibrous dysplasia were followed for an average of 108 months (78 to 260 months). Our patient group consisted of 6 men and 8 women, with a mean age of 13.9 years (2 to 65 years). We reviewed the clinical and pathological features of all 14 patients.

          Results

          Thirteen patients had a lesion in the tibia, while one patient had lesions in both the tibia and the fibula. Initial treatments were observation after biopsy (6 patients), curettage with or without a bone graft (3 patients), resection followed by a free vascularized fibular bone graft (4 patients), or resection and regeneration with the Ilizarov external fixation (1 patient). Curettage was performed on 6 patients due to recurrence or progression after the initial treatment. Among these patients, one was diagnosed with AD from the biopsy of the recurrent lesion. This patient was further treated by segmental resection and pasteurization. After the initial pathology slides of the 13 patients were reviewed with immunohistochemical cytokeratin staining, one patient diagnosis was changed from osteofibrous dysplasia to osteofibrous dysplasia-like adamantinoma.

          Conclusion

          Some patients with osteofibrous dysplasia require close observation because of the high association risk between osteofibrous dysplasia and adamantinoma, Immunohistochemical staining may be helpful in differentiating these two diagnoses.

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          Most cited references31

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          Current trends in the management of adamantinoma of long bones. An international study.

          Adamantinoma of long bones is a rare tumor. Published reviews of the orthopaedic management of adamantinoma have involved limited follow-up of small numbers of patients. The oncological aggressiveness of this tumor is unknown. Limb salvage is currently the treatment of choice for most adamantinomas. The purpose of this study was to evaluate the characteristics of adamantinoma of long bones as well as the oncological outcome and the complications of limb salvage operations. A retrospective study was designed to evaluate the clinical outcomes of limb salvage operations for the treatment of adamantinoma. Data on seventy biopsy-proven cases of adamantinoma treated between 1982 and 1992 at twenty-three different cancer centers in Europe and North America were obtained. The median duration of follow-up was 7.0 years. The male:female ratio was 3:2, and the mean age was thirty-one years. Limb salvage was attempted in 91 percent (sixty-four) of the seventy patients, and the final rate of limb preservation was 84 percent (fifty-nine of seventy). Wide operative margins were obtained in 92 percent (fifty-eight) of sixty-three patients. An intercalary allograft was used to reconstruct the segmental bone defect in 51 percent (thirty-six) of the seventy patients. Reconstruction-related complications occurred in 48 percent (thirty) of sixty-two patients. Nonunion and fracture were the most common complications, occurring in 24 percent (fifteen) and 23 percent (fourteen) of sixty-two patients, respectively. Kaplan-Meier analysis demonstrated a rate of local recurrence of 18.6 percent at ten years. Wide operative margins were associated with a lower rate of local recurrence than marginal or intralesional margins were (p < 0.00005). Kaplan-Meier analysis showed a survival rate of 87.2 percent at ten years. There were no significant relationships between survival and the stage of the tumor (p = 0.058), duration of symptoms (p = 0.90), gender (p = 0.79), or wide operative margins (p = 0.14). Current treatment of adamantinoma, including en bloc tumor resection with wide operative margins and limb salvage, provides lower rates of local recurrence than has been previously reported. In the present study, the limb preservation rate was 84 percent (fifty-nine of seventy), and the survival rate was 87.2 percent at ten years. The rate of complications related to the limb reconstruction was high.
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            Adamantinoma of the long bones. A clinicopathological study of thirty-two patients with emphasis on histological subtype, precursor lesion, and biological behavior.

            The records of thirty-two patients who had had an adamantinoma of the long bones were examined to investigate the relationship between the clinical presentation, the histological subtype, and the method of treatment, and the clinical result. All histological patterns of differentiation that are characteristic of adamantinoma were observed, including the basaloid, spindle-cell, tubular, squamous, and osteofibrous dysplasia-like subtypes. Follow-up data were available for twenty-eight (88 per cent) of the thirty-two patients. These patients were followed for a mean duration of 122 months (range, eleven months to twenty-nine years and two months). Nine patients (32 per cent), all of whom had been managed with an intralesional or marginal procedure, had a local recurrence of the tumor after a mean disease developed in three of the nine patients. In five other patients, metastasis developed without having been preceded by a local recurrence. Thus, the over-all rate of metastasis was 29 per cent (eight patients). The mean duration of survival for the patients who had metastasis was twelve years and eight months. Statistical analysis of various clinicopathological variables revealed intralesional or marginal excision to be the most significant risk factor for a local recurrence or metastasis (p < 0.001). Two patients who had had a presumed osteofibrous dysplasia-like adamantinoma, which contained few isolated keratin-positive epithelial cells within the stroma at the time of presentation, had a full-blown adamantinoma at the time of the local recurrence. Although the clinical course that was observed may be the result of a sampling error, it poses questions as to the regressive nature of osteofibrous dysplasia-like adamantinoma. On the basis of our findings and the data in the literature, we believe that an osteofibrous dysplasia-like adamantinoma may be a precursor lesion of the classic type of adamantinoma.
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              Osteofibrous dysplasia: clinicopathologic study of 80 cases.

              A thorough review of the clinical, radiologic, and histologic features of 80 cases of long bone osteofibrous dysplasia is presented. Of the six cases of osteofibrous dysplasia immunostained with a cytokeratin antibody, two were cytokeratin positive; however, the five cases of fibrous dysplasia that were stained were all negative. Follow-up (mean, 5.4 years; range, 1 month to 31 years) data were available for 41 cases (51%). From the consultation series nine of the 18 patients studied had recurrences, regardless of the different treatment regimens. The average recurrence interval in these patients was 2.7 years. Two patients had incomplete excision and six had biopsy only. All eight of these patients had residual tumors. Among the 16 Mayo Clinic patients, one had recurrence 5 years after the initial treatment. In two cases, 7-year-old and 6-year-old boys, histologic maturation to fibrous dysplasia was observed 4 and 10 years later, respectively. Adamantinoma has not developed in any of the 41 cases of osteofibrous dysplasia for which we have follow-up information. It seems clear that osteofibrous dysplasia does not progress to adamantinoma. Our study suggests that osteofibrous dysplasia is probably a variant of fibrous dysplasia, as demonstrated by the maturation of two lesions of osteofibrous dysplasia to fibrous dysplasia. Surgical treatment may be appropriate in cases with an extensive lesion, pseudoarthrosis, and accentuated tibial bowing. The overall prognosis is good, even with recurrence.
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                Author and article information

                Journal
                Yonsei Med J
                YMJ
                Yonsei Medical Journal
                Yonsei University College of Medicine
                0513-5796
                1976-2437
                30 June 2007
                20 June 2007
                : 48
                : 3
                : 502-510
                Affiliations
                [1 ]Department of Orthopedic Surgery, Yonsei University College of Medicine, Seoul, Korea.
                [2 ]Department of Orthopedic Surgery, Konyang University College of Medicine, Daejeon, Korea.
                [3 ]Department of Pathology, Yonsei University College of Medicine, Seoul, Korea.
                Author notes
                Reprint address: requests to Dr. Soo Bong Hahn, Department of Orthopedic Surgery, Yonsei University College of Medicine, 250 Seongsanno, Seodaemun-gu, Seoul 120-752, Korea. Tel: 82-2-2228-2192, Fax: 82-2-363-1139, sbhahn@ 123456yumc.yonsei.ac.kr
                Article
                10.3349/ymj.2007.48.3.502
                2628088
                17594160
                100e0e89-894d-444e-9f05-d4230ae49611
                Copyright © 2007 The Yonsei University College of Medicine

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                Categories
                Original Article

                Medicine
                osteofibrous dysplasia like adamantinoma,osteofibrous dysplasia,adamantinoma
                Medicine
                osteofibrous dysplasia like adamantinoma, osteofibrous dysplasia, adamantinoma

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