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      Adamantinoma Presenting With Local Recurrence and Inguinal Lymph Node Metastasis: A Case Report

      case-report
      1 , 2 , , 1
      ,
      Cureus
      Cureus
      adamantinoma, local recurrence, metastasis, dedifferentiation, lymph nodes

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          Abstract

          Adamantinoma of long bones is a slow-growing, low-grade primary malignant bone tumor. It is a rare entity and accounts for less than 1% of all primary bone tumors, and in most cases, it occurs in the mid-axis of the tibia of adolescents and young adults. In this report, we describe the case of a 53-year-old woman with a diagnosis of adamantinoma of the left tibia who was initially treated surgically in 2004. Two years later, she presented with local relapse, for which she underwent new surgical management. The patient was referred to our practice 16 years after the initial diagnosis, and she presented with recurrence characterized by ipsilateral inguinal lymph node metastasis. The histological findings and immunohistochemistry were compatible with metastatic adamantinoma with high-grade differentiation, which ultimately required surgical salvage management.

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          Most cited references20

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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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            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 appendicular skeleton--updated.

              An updated review of adamantinoma of the appendicular skeleton now provides 195 well-documented cases from the world literature. An additional five new cases are added. Statistically, the tumor remains unusually prevalent in the tibia, but all other major limb bones have been involved, and involvement of several short bones rarely has been reported. The neoplasm is more commonly found in males, but higher earlier age incidence is found in females. The frequent history of preceding trauma may indeed be important in tumor formation. The histogenesis of the tumor is now considered to be epithelial in origin by ultrastructural and immunohistochemical methods. A high incidence of recurrence or metastases is found with inadequate cancer surgery. Known mortalities have indicated severe metastatic disease by aggressive-appearing cells. Previously, early amputation had provided good results, but wide excision or segmental resection with grafting techniques are equally successful. The recent work with allograft replacement of a widely excised segment has shown good early results.
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                Author and article information

                Journal
                Cureus
                Cureus
                2168-8184
                Cureus
                Cureus (Palo Alto (CA) )
                2168-8184
                19 February 2021
                February 2021
                : 13
                : 2
                : e13439
                Affiliations
                [1 ] Clinical Oncology, Universidad el Bosque/Instituto Nacional de Cancerología, Bogota, COL
                [2 ] General Medicine, Universidad del Magdalena/Instituto Nacional de Cancerología, Bogota, COL
                Author notes
                Eduardo Luis Canales Pacheco edlucan@ 123456hotmail.com
                Article
                10.7759/cureus.13439
                7981846
                78680a1f-c482-4d42-ab51-843601ec313b
                Copyright © 2021, Zapata Laguado et al.

                This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                History
                : 18 February 2021
                Categories
                Internal Medicine
                Radiation Oncology
                Orthopedics

                adamantinoma,local recurrence,metastasis,dedifferentiation,lymph nodes

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