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      Adamantinoma of tibia: excision and reconstruction with free vascularized fibula flap: a case report and review of literature

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          Abstract

          Adamantinoma is a rare malignant tumour usually affecting the diaphysis of long bones. The tumour most commonly affects the tibia. Conventional management involves excision with a wide margin, reconstruction and sometimes amputation. Multiple options are available, but reconstruction depends upon the size of the bony defect and available resources. None has proved to be the favourable one. We have analysed the advantages and shortcomings of various methods used. In our case, a 33-year-old male patient presented with a large adamantinoma of the midtibial region of the left leg, which was managed with excision and reconstruction of long segment bony defect with free vascularized osteocutaneous fibula flap. There are osseointegration and hypertrophy of the vascularized bone with good functional gain in long term follow-up. Autologous bone reconstruction after adamantinoma excision with microvascular free fibula flap in large bone segment defects salvage the limb with satisfactory functional outcome.

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          Most cited references8

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          Bone tumors and tumorlike conditions: analysis with conventional radiography.

          The approach to the radiographic diagnosis of bone tumors consists of analyzing the lesion in an organized fashion, with attention to the specific radiographic features of tumor location, margins, and zone of transition; periosteal reaction; mineralization; size and number of lesions; and presence of a soft-tissue component. Patient age is also an important clinical factor in the diagnosis of bone tumors, because various lesions have predilections for specific age groups. (c) RSNA, 2008.
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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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              Free vascularised fibular grafting for reconstruction after tumour resection.

              We have reviewed 30 patients at a mean of 36 months after free vascularised fibular transfer to reconstruct massive skeletal defects after resection of primary bone tumours. There were 23 malignant and 7 benign neoplasms, half in the lower limb and half in the upper. Arthrodesis was performed in 15 and intercalary bone replacement in 15. The mean fibular graft length was 189 mm. Union was achieved in 27 (90%) at an average of 7.6 months, and the 3-year survival was 89%. There was a high complication rate (50%), but most resolved without greatly influencing the final outcome. There was local recurrence in two (6.7%), but 16 of the 24 assessed patients (67%) had satisfactory functional results. This is a reasonably effective means of reconstruction for limb salvage after resection of tumours.
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                Author and article information

                Contributors
                Journal
                J Surg Case Rep
                J Surg Case Rep
                jscr
                Journal of Surgical Case Reports
                Oxford University Press
                2042-8812
                April 2022
                15 April 2022
                15 April 2022
                : 2022
                : 4
                : rjac165
                Affiliations
                Department of Burns & Plastic Surgery , All India Institute of Medical Sciences , Raipur, Chhattisgarh, India
                Department of Burns & Plastic Surgery , All India Institute of Medical Sciences , Raipur, Chhattisgarh, India
                Department of Orthopedic Surgery , All India Institute of Medical Sciences , Raipur, Chhattisgarh, India
                Department of General Surgery , All India Institute of Medical Sciences , Raipur, Chhattisgarh, India
                Department of Burns & Plastic Surgery , All India Institute of Medical Sciences , Raipur, Chhattisgarh, India
                Author notes
                Correspondence address. Department of Burns & Plastic Surgery, First Floor, D1 Block, All India Institute of Medical Sciences, Raipur, Chhattisgarh 492099, India. Tel: +91-9437616743; E-mail: mishra.jitenkumar@ 123456gmail.com
                Article
                rjac165
                10.1093/jscr/rjac165
                9013241
                a08a82ac-39e4-480b-aa80-5c20887f4f4c
                Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2022.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( https://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

                History
                : 7 February 2022
                : 27 March 2022
                Page count
                Pages: 4
                Categories
                Case Report
                AcademicSubjects/MED00910
                jscrep/0130

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