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1.
Am J Case Rep ; 25: e941248, 2024 Mar 15.
Artigo em Inglês | MEDLINE | ID: mdl-38486378

RESUMO

BACKGROUND Adamantinoma is a rare low-grade malignant bone tumor, usually found in the tibial diaphysis and metaphysis, with histological similarities to mandibular ameloblastoma. The most effective treatment of recurrent adamantinoma is not yet clear. This report is of a 22-year-old woman with recurrent tibial adamantinoma treated with the tyrosine kinase inhibitor pazopanib. CASE REPORT We report the case of a 22-year-old woman who was referred to our center for a suspicious bone lesion in the right tibia. Bone biopsy findings were consistent with an adamantinoma. En bloc resection was completed successfully, with no postoperative complications. Five years later, a positive emission tomography scan revealed mildly increased tracer uptake near the area of the previous lesion and in the right inguinal lymph node. Biopsies of the lesion and inguinal lymph node confirmed recurrence of the adamantinoma. Due to abdominal and pelvic metastasis, the patient underwent surgical debulking, along with an appendectomy, right salpingo-oophorectomy, intraoperative radiation therapy, and hyperthermic intraperitoneal chemotherapy. Subsequently, the patient was placed on pazopanib for 4 months; however, her tumor continued to worsen after 4 months of chemotherapy. Currently, the patient is receiving gemcitabine and docetaxel as second-line medical therapy. CONCLUSIONS This report showed that pazopanib as standalone treatment does not appear to have promising role on patient outcomes. To the best of our knowledge, this is the second report of pazopanib in the treatment of adamantinoma.


Assuntos
Adamantinoma , Ameloblastoma , Neoplasias Ósseas , Indazóis , Pirimidinas , Sulfonamidas , Feminino , Humanos , Adulto Jovem , Adamantinoma/patologia , Adamantinoma/secundário , Adamantinoma/cirurgia , Ameloblastoma/complicações , Ameloblastoma/patologia , Ameloblastoma/cirurgia , Neoplasias Ósseas/patologia , Tíbia/cirurgia
3.
Bone Joint J ; 99-B(3): 409-416, 2017 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-28249983

RESUMO

AIMS: The aim of this study was to identify any progression between benign osteofibrous dysplasia (OFD), OFD-like adamantinoma and malignant adamantinoma, and to investigate the rates of local recurrence, metastases and survival, in order to develop treatment algorithms for each. PATIENTS AND METHODS: A single institution retrospective review of all patients presenting with OFD, OFD-like adamantinoma and adamantinoma between 1973 and 2012 was undertaken. Complete data were available for 73 patients (42 with OFD; ten with an OFD-like adamantinoma and 21 with an adamantinoma). The mean follow-up was 10.3 years (3 to 25) for OFD, 9.2 years (3.0 to 26.3) for OFD-like and 11.6 years (0.25 to 33) for adamantinoma. RESULTS: The mean age at diagnosis for OFD was 13.5 years (1 to 49), 10.5 years (6 to 28) for OFD-like and 34 years (14 to 86) for adamantinoma. A total of 24 of the 42 patients with OFD (57%) have not required any treatment and have been managed with observation. A total of 18 of the 42 patients with OFD underwent surgery, 13 with curettage and five with resection. In all, three patients developed recurrence following curettage (23%) but none following resection. All these patients were cured with further limited surgery. A total of six patients initially diagnosed with OFD were subsequently found to have OFD-like adamantinoma. Of the ten patients initially diagnosed with OFD-like adamantinoma, three (30%) were managed with observation alone and seven underwent surgery, two with curettage and five with resection. Local recurrence arose in two patients, one each after curettage and resection. No patients with either OFD or an OFD-like adamantinoma developed metastases or had progression to adamantinoma. All patients with an adamantinoma were treated by surgery, three with curettage, six with amputation and 12 with excision. In all, two of the three treated with curettage developed local recurrence, requiring further surgery. Late development of both local recurrence and metastases led to a ten year disease specific survival of 93% which had dropped to 39% by 20 years. CONCLUSION: We found no evidence of progression from OFD to adamantinoma. Conservative management with observation or curettage is often successful for patients with OFD and OFD-like adamantinoma. Resection with clear margins is required for patients with adamantinoma. Late tumour recurrence is not uncommon in adamantinoma and prolonged follow-up should be considered. Cite this article: Bone Joint J 2017;99-B:409-16.


Assuntos
Adamantinoma/diagnóstico , Doenças do Desenvolvimento Ósseo/diagnóstico , Adamantinoma/secundário , Adamantinoma/terapia , Adolescente , Doenças do Desenvolvimento Ósseo/terapia , Transformação Celular Neoplásica , Criança , Pré-Escolar , Progressão da Doença , Feminino , Seguimentos , Humanos , Lactente , Estimativa de Kaplan-Meier , Masculino , Recidiva Local de Neoplasia , Radiografia , Estudos Retrospectivos , Adulto Jovem
4.
Gan To Kagaku Ryoho ; 42(12): 1677-9, 2015 Nov.
Artigo em Japonês | MEDLINE | ID: mdl-26805135

RESUMO

This case involved a 28-year-old man who had undergone surgery and perioperative chemotherapy for an adamantinoma of the right tibia with multiple lung metastases. Sixteen months after the initial diagnosis, CT revealed an 8 cm diameter liver metastasis and right pneumothorax with little change in the lung metastases. Liver resection and partial pneumonectomy were performed. Pathologic findings confirmed that both liver and lung specimens had metastases from the adamantinoma. Dissimilar from the primary lesion with much interstitial tissue and spindle-shaped cells, the liver metastasis had very dense cell proliferation without interstitial tissue and dominant epithelial parts, suggesting a higher malignant potential. If other lesions are under good control, resection of the newly appearing metastasis, which has a higher malignant potential, might improve prognosis. Further accumulation of cases and detailed studies is required.


Assuntos
Adamantinoma/tratamento farmacológico , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Neoplasias Ósseas/tratamento farmacológico , Neoplasias Ósseas/patologia , Neoplasias Hepáticas/tratamento farmacológico , Neoplasias Pulmonares/tratamento farmacológico , Adamantinoma/secundário , Adamantinoma/cirurgia , Adulto , Neoplasias Ósseas/cirurgia , Hepatectomia , Humanos , Neoplasias Hepáticas/secundário , Neoplasias Hepáticas/cirurgia , Neoplasias Pulmonares/secundário , Neoplasias Pulmonares/cirurgia , Masculino , Pneumonectomia , Resultado do Tratamento
5.
J Orthop Surg (Hong Kong) ; 22(3): 420-2, 2014 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-25550030

RESUMO

Adamantinoma is a rare, low-grade, malignant bone tumour. We report on a 46-year-old woman who had early multiple recurrences of adamantinoma of the right tibia and late metastasis to the lung and ribs 13 years after the first surgical treatment. She underwent multiple complete tumour excisions and eventually below-knee amputation and removal of the left lung and sixth to eighth ribs.


Assuntos
Adamantinoma/cirurgia , Neoplasias Ósseas/cirurgia , Neoplasias Pulmonares/cirurgia , Recidiva Local de Neoplasia/cirurgia , Tíbia/cirurgia , Adamantinoma/secundário , Amputação Cirúrgica , Neoplasias Ósseas/patologia , Feminino , Humanos , Neoplasias Pulmonares/secundário , Pessoa de Meia-Idade , Pneumonectomia , Costelas/cirurgia
7.
Clin Imaging ; 35(6): 483-7, 2011.
Artigo em Inglês | MEDLINE | ID: mdl-22040797

RESUMO

Adamantinoma is a rare primary bone malignancy with a predilection for the tibial cortex. Metastases have been reported to the long bones, lung, pleura and spine. We present a 26-year-old pregnant woman with metastatic disease to the posterior cul-de-sac, lungs, liver and retroperitoneum, which collectively have not been previously reported in a single patient to our knowledge.


Assuntos
Adamantinoma/secundário , Neoplasias Ósseas/patologia , Escavação Retouterina , Neoplasias Peritoneais/secundário , Complicações Neoplásicas na Gravidez , Tíbia , Adulto , Feminino , Humanos , Neoplasias Hepáticas/secundário , Neoplasias Pélvicas/secundário , Gravidez
8.
Ann Diagn Pathol ; 15(5): 347-54, 2011 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-20952283

RESUMO

Adamantinoma is a rare neoplasm that characteristically involves the tibia. In many instances, typical location within the tibia, very slow course, and a typical radiographic appearance can strongly suggest the correct diagnosis. We present a case that has both unusual radiographic findings and uncharacteristic histology. In this case, radiologic imaging showed a poorly defined lytic lesion within the distal, lateral tibia extending to the joint with central necrosis, overlying periosteal reaction and possible tumor spread into soft tissue. The histology of this lesion showed pronounced vascularity and surrounding large neoplastic cells with plasmacytoid morphology. The combination of these features led to an initial misdiagnosis as metastatic carcinoma from unknown primary.


Assuntos
Adamantinoma , Tíbia/patologia , Adamantinoma/diagnóstico por imagem , Adamantinoma/patologia , Adamantinoma/secundário , Adulto , Biomarcadores Tumorais/análise , Carcinoma/diagnóstico , Carcinoma/metabolismo , Carcinoma/patologia , Diagnóstico Diferencial , Neoplasias Gastrointestinais/diagnóstico , Neoplasias Gastrointestinais/metabolismo , Neoplasias Gastrointestinais/patologia , Humanos , Imuno-Histoquímica , Masculino , Plasmócitos/patologia , Radiografia
9.
J Bras Pneumol ; 34(6): 425-9, 2008 Jun.
Artigo em Português | MEDLINE | ID: mdl-18622511

RESUMO

Here, we describe two cases of lung metastasis of adamantinoma of long bones, a low-grade bone neoplasm that rarely metastasizes. In both cases, the clinical presentation of the metastases was characterized by spontaneous pneumothorax secondary to tumor cavitation, a phenomenon described in only three of the studies reviewed in the literature. Clinical, radiological, and anatomopathological findings, as well as the procedures adopted in the two cases, are described.


Assuntos
Adamantinoma/secundário , Neoplasias Ósseas/patologia , Neoplasias Pulmonares/secundário , Pneumotórax/etiologia , Adulto , Humanos , Masculino , Pessoa de Meia-Idade
10.
J. bras. pneumol ; 34(6): 425-429, jun. 2008. ilus
Artigo em Inglês, Português | LILACS | ID: lil-485904

RESUMO

Descrevem-se dois casos de metástases pulmonares de adamantinoma de ossos longos, o qual é uma neoplasia óssea de baixo grau que raramente metastatiza. Nos dois casos a apresentação clínica das metástases se deu por pneumotórax espontâneo secundário a escavação tumoral, fenômeno descrito em apenas três dos trabalhos consultados na literatura. São descritos os achados clínicos, radiológicos e anatomopatológicos, bem como os procedimentos adotados nos dois casos.


Here, we describe two cases of lung metastasis of adamantinoma of long bones, a low-grade bone neoplasm that rarely metastasizes. In both cases, the clinical presentation of the metastases was characterized by spontaneous pneumothorax secondary to tumor cavitation, a phenomenon described in only three of the studies reviewed in the literature. Clinical, radiological, and anatomopathological findings, as well as the procedures adopted in the two cases, are described.


Assuntos
Adulto , Humanos , Masculino , Pessoa de Meia-Idade , Adamantinoma/secundário , Neoplasias Ósseas/patologia , Neoplasias Pulmonares/secundário , Pneumotórax/etiologia
11.
J Bone Joint Surg Br ; 89(3): 388-9, 2007 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-17356156

RESUMO

Adamantinoma is a rare tumour of long bones that occurs most commonly in the tibia. Its pathogenesis is unknown. It is locally aggressive and recurrences are common after resection. Metastases have been reported in 10% to 20% of cases, most commonly in the lungs and rarely in the lymph nodes. We report a patient who developed a skeletal metastasis four years after resection of the primary tumour. There was no evidence of recurrence at the primary site or of secondary deposits in the lungs.


Assuntos
Adamantinoma/secundário , Neoplasias Ósseas/patologia , Neoplasias Femorais/secundário , Tíbia , Adamantinoma/patologia , Adamantinoma/cirurgia , Adolescente , Neoplasias Ósseas/cirurgia , Neoplasias Femorais/patologia , Humanos , Masculino , Tíbia/patologia , Tíbia/cirurgia
12.
Acta Cytol ; 50(5): 567-73, 2006.
Artigo em Inglês | MEDLINE | ID: mdl-17017449

RESUMO

BACKGROUND: Adamantinoma is a rare primary bone neoplasm of low malignant potential that may recur or metastasize in a mall percentage of patients. The myriad histologic patterns may cause difficulty in distinguishing this tumor from other primary or metastatic neoplasms. The cytomorphologic findings of fine needle aspiration biopsy were reported previously in only a small number of cases. CASE: A 32-year-old man presented with a mass in the distal side of the left leg that was diagnosed as classic adamantinoma by open biopsy. Local recurrence and pulmonary metastases were confirmed by fine needle aspiration biopsy, which showed low grade, uniform cells with nuclear membrane grooves. The patient underwent a below-the-knee amputation and is receiving palliative treatment for progressive pulmonary spread. CONCLUSION: The diagnosis of adamantinoma requires knowledge of compatible clinical and radiologic studies as well as understanding of the variable histologic patterns that one may encounter. Fine needle aspiration biopsy is particularly useful in the diagnosis of recurrent and metastatic adamantinoma. This case report describes a distinctive cytomorphologic feature of nuclear grooves that may be a useful aid in distinguishing the tumor cells of adamantinoma from other cell types.


Assuntos
Adamantinoma/diagnóstico , Neoplasias Ósseas/diagnóstico , Neoplasias Pulmonares/diagnóstico , Pulmão/patologia , Recidiva Local de Neoplasia/diagnóstico , Tíbia/patologia , Adamantinoma/secundário , Adamantinoma/cirurgia , Adulto , Amputação Cirúrgica , Biomarcadores Tumorais/análise , Biomarcadores Tumorais/biossíntese , Biópsia por Agulha Fina , Neoplasias Ósseas/patologia , Neoplasias Ósseas/cirurgia , Forma Celular , Diagnóstico Diferencial , Progressão da Doença , Células Epiteliais/patologia , Humanos , Neoplasias Pulmonares/secundário , Neoplasias Pulmonares/cirurgia , Imageamento por Ressonância Magnética , Masculino , Recidiva Local de Neoplasia/cirurgia , Membrana Nuclear/patologia , Prognóstico , Tíbia/cirurgia
13.
Skeletal Radiol ; 35(3): 190-3, 2006 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-16402219

RESUMO

A 26-year-old female with a tibial lesion diagnosed as an adamantinoma was treated with intra-lesional curettage, bone grafting and intra-medullary nailing. Six years post-surgery, she presented with an asymptomatic primary site but with a metastatic lesion in the mid-shaft of the ipsilateral femur and lung metastases. The femoral lesion was treated with wide excision and reconstructed with an allograft and plate fixation. Pulmonary metastatectomy was carried out for the lung lesions. A follow-up CT scan of the chest at 1 year after the surgery for the metastatic lesions revealed fresh unresectable bilateral metastases. Although cases of local recurrences and pulmonary metastases in adamantinoma are reported, this case is unusual in presenting without a local recurrence but with simultaneous skeletal and pulmonary metastases.


Assuntos
Adamantinoma/secundário , Neoplasias Ósseas/patologia , Neoplasias Femorais/secundário , Neoplasias Pulmonares/secundário , Tíbia , Adamantinoma/diagnóstico por imagem , Adamantinoma/cirurgia , Adulto , Neoplasias Ósseas/diagnóstico por imagem , Neoplasias Ósseas/cirurgia , Feminino , Neoplasias Femorais/diagnóstico por imagem , Humanos , Neoplasias Pulmonares/diagnóstico por imagem , Cintilografia , Tomografia Computadorizada por Raios X
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