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          Adamantinoma accounts for less than 1% of the primary bone neoplasms. The tibia is the most affected bone and it is predominant in male patients between the second and third decades of life. The objective of this study is to obtain epidemiological and clinical information on patients with adamantinoma of the tibia treated surgically between 1989 and 2016.


          Retrospective series of seven patients diagnosed with adamantinoma of the tibia that underwent surgery at the orthopedic oncology service of our hospital. The information was obtained from the medical records and histopathological reports of our institution.


          A total of 2870 medical records with histological reports were evaluated. Seven cases of adamantinoma of the tibia were included. The mean age was 28.5 (17-49) years. We found a predominance of females (71.4%) and the most affected side was the left one, with four cases (57.1%). The biopsy revealed bone adamantinoma in four (57.1%) patients, while the diagnosis of the other patients was confirmed after the histological examination of the surgical specimen. All the patients underwent surgery as definitive treatment. No positive margins were reported. No local recurrence (LR) was reported and two patients had distant metastasis (DM).


          The prognosis of survival in cases of adamantinoma of the tibia is high. The rates of LR and DM were low. Surgical treatment with extensive tumor resection is the treatment of choice. Level of Evidence IV, Case Series.



          O adamantinoma representa menos de 1% das neoplasias ósseas primárias. Afeta predominantemente a tíbia, em pacientes do sexo masculino entre a segunda e terceira décadas da vida. O objetivo deste trabalho é obter informação epidemiológica e clínica dos pacientes com adamantinoma da tíbia, tratados mediante cirurgia entre 1989 e 2016.


          Série retrospectiva de sete pacientes com diagnóstico de adamantinoma da tíbia, tratados cirurgicamente no serviço de oncologia ortopédica do nosso hospital. A informação foi obtida dos relatos clínicos e patológicos do instituto.


          Um total de 2870 prontuários com relatos anatomopatológicos foram revisados. Sete casos de pacientes com adamantinoma na tíbia foram encontrados. A média de idade foi de 28,5 anos (17-49). Encontramos predominância do sexo feminino (71,4%). O lado mais afetado foi o esquerdo, com quatro (57,1%) casos. A biópsia diagnosticou adamantinoma em 57,1% dos casos e o diagnóstico dos outros casos foi definido após exame da peça cirúrgica. Todos os pacientes receberam tratamento cirúrgico como terapia definitiva. Não foram reportadas margens comprometidas. Nenhum paciente apresentou recorrência local (RL). Dois pacientes apresentaram metástase à distância (MD).


          O prognóstico de sobrevida do adamantinoma da tíbia é alto. Apresenta taxas baixas de RL e MD. A cirurgia com ampla ressecção do tumor é o tratamento de escolha. Nível de Evidência IV, Série de Casos.

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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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              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.

                Author and article information

                Acta Ortop Bras
                Acta Ortop Bras
                Acta Ortopedica Brasileira
                ATHA EDITORA
                : 26
                : 4
                : 252-254
                [1 ]Orthopedic Oncology Group, Instituto de Ortopedia, Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de São Paulo, São Paulo, SP, Brazil.
                [2 ]Instituto de Ortopedia, Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de São Paulo, São Paulo, SP, Brazil.
                [3 ]Department of Orthopedics and Traumatology, Faculdade de Medicina, Universidade de São Paulo, São Paulo, SP, Brazil.
                Author notes
                Correspondence: Juan Pablo Zumárraga. Rua Ovídio Pires de Campos, 333, Cerqueira Cesar, São Paulo, SP, Brazil. 05403-010. juanpzumarraga@ 123456hotmail.com

                All authors declare no potential conflict of interest related to this article.

                AUTHORS’ CONTRIBUTIONS: Each author made significant individual contributions to this manuscript. JPZ (0000-0001-5941-7714)*, RC (0000-0002-7853-1325)*, MTK (000-0003-3384-7905)*, AMB (0000-0002-0830-4602)*, FGS (0000-0002-6854-6072)*, and OPC (0000-0002-1128-7292)*, were the main contributors in writing this work. JPZ participated in: research, design, writing and data collection; RC participated in: concept, research, writing and data collection; MTK participated in: concept, research, writing and data collection; AMB participated in: concept, design, and analysis; FGS participated in: research, design and data collection; OPC participated in: concept, writing and analysis. *ORCID (Open Researcher and Contributor ID).


                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 work is properly cited.

                Page count
                Figures: 1, Tables: 1, Equations: 0, References: 23, Pages: 3
                Original Article

                adamantinoma,tibia,bone neoplasms,biopsy,surgical oncology,tíbia,neoplasias ósseas,biópsia,oncologia cirúrgica


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