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      Epidemiological Study of Adamantinoma from US Surveillance, Epidemiology, and End Results Program: III Retrospective Analysis

      research-article
      1 , 2 , , 3
      Journal of Oncology
      Hindawi

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          Abstract

          Objective

          Adamantinomas are rare low-grade malignant bone tumors. This study aims to describe the demographic characteristics and survival rates of patients suffering from adamantinomas.

          Methods

          The National Institute of Cancer Surveillance, Epidemiology, and Recent Results (SEER) database was used, and patients diagnosed with adamantinoma between 1973 and 2016 were screened. Patients were classified according to sex, age, race/ethnicity, and marital status, and also tumors were classified according to year of diagnosis, laterality, type of treatment, and follow-up.

          Results

          The mean age of patients was 30.8 ± 16.7 (range: 4–75). A total of 92 patients were identified; of these, 43 were females and 49 were males. The mean follow-up period was 138.1 ± 90.3 (range: 1–156) months. Mean survival duration was 287.8 ± 15.4 (95% CI: 257.7–317.9) months. Five- and ten-year survival rates were 98.8% and 91.5%, respectively. Besides, survival time was also observed to be independent of gender, age groups, race, marital status, tumor location, and year of diagnosis.

          Conclusion

          Adamantinoma is a very rare bone tumor that affects the long bones in lower extremities and is more common in men. Five- and 10-year survival prognoses are reasonably satisfactory. Also, survival time is independent of variables such as gender, age, and tumor location.

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          Most cited references19

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          Adamantinoma: A clinicopathological review and update

          Adamantinoma is a primary low-grade, malignant bone tumor that is predominantly located in the mid-portion of the tibia. The etiology of the tumor is still a matter of debate. The initial symptoms of adamantinoma are often indolent and nonspecific and depend on location and extent of the disease. Histologically, classic adamantinoma is a biphasic tumor characterized by epithelial and osteofibrous components that may be intermingled with each other in various proportions and differentiating patterns. To assure the histological diagnosis, pathologists should employ immunohistochemistry for demonstrating the sometimes sparse epithelial cell nests when the radiological features are suggestive of adamantinoma. There is paucity of compiled data over adamantinoma in the literature, hence authors tried to make a comprehensive review which must be of use to beginners and trained pathologists. Our objective is to further define the clinicoradiologic features and pathologic spectra of adamantinoma.
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            Adamantinoma of long bones. A clinicopathologic study of 85 cases.

            A study of 85 adamantinomas of long bones revealed that 70 were in the tibia (11 of which also involved the fibula), six were in the femur, three were in the ulna, two were in the humerus, two were in the fibula, one was in the radius, and one arose in the soft tissue anterior to the tibia. Most patients presented with pain and swelling and were aged 10 to 30 years. The histologic appearance was that of epithelial islands in a fibrous stroma, usually with a prominent vascular pattern and a transition between the two. Twenty-six (31%) patients had recurrent local disease, 13 (15%) developed lung metastasis, and six (7%) had lymph node metastasis. Nine patients with lung metastasis had preceding recurrent local disease. Risk factors for recurrent or metastatic disease included male sex, pain, symptoms of less than 5 years' duration, and initial treatment by biopsy, curettage, excision, or resection. The only histologic feature associated with an increased recurrence rate was lack of squamous differentiation. Eleven patients died of their disease, and seven are alive with metastatic disease. Forty-one patients were still alive without disease 1 month to 47 years after treatment. Our results indicate that amputation or, when technically feasible, wide en bloc resection is the treatment of choice. All patients require long-term follow-up for evidence of local recurrence or lung metastasis.
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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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                Author and article information

                Contributors
                Journal
                J Oncol
                J Oncol
                JO
                Journal of Oncology
                Hindawi
                1687-8450
                1687-8469
                2020
                16 June 2020
                : 2020
                : 2809647
                Affiliations
                1Department of Orthopedics and Traumatology, Ankara Yildirim Beyazit University, Ankara, Turkey
                2Department of Orthopedics and Traumatology, Dr Abdurrahman Yurtaslan Oncology Training and Research Hospital, Ankara 06200, Turkey
                3Department of Orthopaedics, Rutgers New Jersey Medical School, 140 Bergen Street, Suite D, Newark, NJ 07103, USA
                Author notes

                Academic Editor: San-Lin You

                Author information
                https://orcid.org/0000-0003-4344-6742
                https://orcid.org/0000-0002-6753-9321
                https://orcid.org/0000-0002-9114-1890
                Article
                10.1155/2020/2809647
                7315319
                e58addcb-9c12-42be-883e-7ac6a8e244aa
                Copyright © 2020 Mahmut Nedim Aytekin et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 6 March 2020
                : 4 May 2020
                : 21 May 2020
                Categories
                Research Article

                Oncology & Radiotherapy
                Oncology & Radiotherapy

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