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      Osteofibrous dysplasia-like adamantinoma treated via intercalary segmental resection with partial cortex preservation using pedicled vascularized fibula graft: a case report

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          Abstract

          Background

          Morphologically, osteofibrous dysplasia-like adamantinoma is thought to be intermediate between osteofibrous dysplasia and adamantinoma. Its treatment is not well established owing to its rarity.

          Case presentation

          We report about of a 10-year-old girl with osteofibrous dysplasia-like adamantinoma initially diagnosed as osteofibrous dysplasia and treated via intercalary segmental resection with partial cortex preservation using a pedicled vascularized fibula graft for reconstruction. Bone union was observed 9 weeks after surgery. Twenty-two months after the definitive surgery, no recurrence was observed.

          Conclusion

          This case illustrates the upgrade from osteofibrous dysplasia to osteofibrous dysplasia-like adamantinoma. The surgical method may aid the treatment of osteofibrous dysplasia-like adamantinoma with incomplete cortex involvement of the tumor.

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          Most cited references8

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          Osteofibrous dysplasia: clinicopathologic study of 80 cases.

          Steven McLeod, Christopher Y. Park, John Pritchard … (1993)
          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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            Osteofibrous dysplasia and adamantinoma.

            J Möst, Carrie Y Inwards, Jae H. Sim (2010)
            Osteofibrous dysplasia (OFD) is a rare, benign, fibro-osseous lesion that typically is seen within the cortex of the tibia in children. Adamantinoma (AD) is a rare, low-grade malignant primary bone tumor that occurs most often in the tibia and/or fibula of adolescent persons and young adults; however, it has been reported in other long bones, as well. Immunohistochemical and ultrastructural evidence has shown that the neoplastic cell in AD derives from an epithelial lineage. More recently, published reports have described another clinical entity-differentiated or OFD-like AD-that appears to lie between OFD and AD along a spectrum of disease. Controversy exists as to whether OFD is a precursor lesion to AD or whether OFD may be a residual lesion resulting from a spontaneously regressing AD. Management of OFD varies from observation to surgical intervention, depending on the age of the patient and the extent of the lesion. Management of AD requires surgical resection with wide margins, followed by appropriate reconstruction, to minimize the risk of local recurrence or metastasis.
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              Osteofibrous dysplasia and adamantinoma in children and adolescents: a clinicopathologic reappraisal.

              B Liegl-Atzwanger, C Gebhardt, Briana C Gleason … (2008)
              Osteofibrous dysplasia (OFD) and adamantinoma are rare and most commonly arise in the tibia of young individuals. Although OFD has typical histopathologic features, areas resembling OFD have often been noted at the periphery of otherwise classic adamantinomas, and some have suggested that OFD may be either a precursor to or a regressive phase of adamantinoma. The so-called OFD-like adamantinoma encompasses some features of both OFD and adamantinoma. We studied the clinical, imaging, histopathologic, immunohistochemical, ultrastructural, and molecular features of 16 OFD and 8 adamantinomas (5 OFD-like and 3 classic) in an attempt to further define their morphology, clinical course, and relationship. Patients with OFD were generally younger than those with adamantinoma. Osteoblastic and osteoclastic activity was more prominent in OFD than in OFD-like adamantinoma. In addition to the inconspicuous small clusters of epithelial cells in OFD-like adamantinoma, isolated keratin-positive cells with a unique ultrastructural hybrid fibroblastic-epithelial phenotype were found in the stroma of all OFD and OFD-like adamantinomas. Fluorescence in situ hybridization analysis revealed trisomies 7, 8, and/or 12 in the spindle cell stroma of OFD, OFD-like, and classic adamantinoma, supporting a neoplastic origin of OFD and a common histogenesis for all 3 lesions. Trisomies were not observed in osteoblasts or osteoclasts suggesting that the osseous component is reactive and non-neoplastic. Of the 11 OFD patients with follow-up (median, 4.5 y), all 3 who underwent incisional biopsy had persistent, nonprogressive disease and 2 of 8 who underwent curettage or wide excision had recurrence; none developed adamantinoma. All 6 adamantinoma patients with follow-up (3 classic and 3 OFD-like) were treated with wide excision. One with classic adamantinoma died of pulmonary metastases 9 years after presentation; the other 5 were free of disease with a median follow-up of 12 years. None of the classic adamantinomas evolved into OFD-like adamantinoma or OFD. Although the histopathology, immunohistochemistry, ultrastructure, and cytogenetics indicate that these lesions are closely related, our data and the literature suggest that only classic adamantinoma has malignant potential. OFD, OFD-like adamantinoma, and classic adamantinoma appear to show a progressive complexity of cytogenetic aberrations, perhaps indicative of a multistep neoplastic transformation.
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                Author and article information

                Contributors
                Makoto Emori:
                ORCID: http://orcid.org/0000-0003-2512-2795
                emrmkt@yahoo.co.jp
                Journal
                Journal ID (nlm-ta): World J Surg Oncol
                Journal ID (iso-abbrev): World J Surg Oncol
                Title: World Journal of Surgical Oncology
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1477-7819
                Publication date (Electronic): 13 August 2020
                Publication date PMC-release: 13 August 2020
                Publication date Collection: 2020
                Volume: 18
                Electronic Location Identifier: 203
                Affiliations
                [1 ]GRID grid.263171.0, ISNI 0000 0001 0691 0855, Department of Orthopedic Surgery, , Sapporo Medical University School of Medicine, ; West 16, South 1, Chuo- ku, Sapporo, Hokkaido 060-8543 Japan
                [2 ]GRID grid.263171.0, ISNI 0000 0001 0691 0855, Department of Diagnostic Pathology, , Sapporo Medical University School of Medicine, ; Sapporo, Hokkaido Japan
                Author information
                http://orcid.org/0000-0003-2512-2795
                Article
                Publisher ID: 1983
                DOI: 10.1186/s12957-020-01983-9
                PMC ID: 7427073
                PubMed ID: 32792007
                SO-VID: b3e70c11-3439-4d92-a59a-1ef4639fe7bb
                Copyright © © The Author(s) 2020
                License:

                Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

                History
                Date received : 20 May 2020
                Date accepted : 31 July 2020
                Funding
                Funded by: JSPS KAKENHI
                Award ID: 17K10974
                Award Recipient :
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2020

                ScienceOpen disciplines: Surgery
                Keywords: osteofibrous dysplasia,vascularized fibula graft,bone tumor
                Data availability:
                ScienceOpen disciplines: Surgery
                Keywords: osteofibrous dysplasia, vascularized fibula graft, bone tumor

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