Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to Denosumab

Giant cell tumor of bone (GCT) is a rare, locally aggressive neoplasm characterized by the presence of giant cells with osteoclast activity. Its biology involves the overexpression of the Receptor Activator of Nuclear Factor kB Ligand (RANKL) by osteoclast-like giant cells and tumor stromal cells, w...

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Main Authors: Natalia M. P. Fraile, Diego Toloi, Ceci O. Kurimori, Adriana R. B. Matutino, Alberto Codima, Veridiana P. Camargo, Olavo Feher, Rodrigo R. Munhoz
Format: Article
Language:English
Published: Wiley 2015-01-01
Series:Case Reports in Oncological Medicine
Online Access:http://dx.doi.org/10.1155/2015/626741
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author Natalia M. P. Fraile
Diego Toloi
Ceci O. Kurimori
Adriana R. B. Matutino
Alberto Codima
Veridiana P. Camargo
Olavo Feher
Rodrigo R. Munhoz
author_facet Natalia M. P. Fraile
Diego Toloi
Ceci O. Kurimori
Adriana R. B. Matutino
Alberto Codima
Veridiana P. Camargo
Olavo Feher
Rodrigo R. Munhoz
author_sort Natalia M. P. Fraile
collection DOAJ
description Giant cell tumor of bone (GCT) is a rare, locally aggressive neoplasm characterized by the presence of giant cells with osteoclast activity. Its biology involves the overexpression of the Receptor Activator of Nuclear Factor kB Ligand (RANKL) by osteoclast-like giant cells and tumor stromal cells, which has been shown to be an actionable target in this disease. In cases amenable to surgical resection, very few therapeutic options were available until the recent demonstration of significant activity of the anti-RANK-ligand monoclonal antibody denosumab. Here we present a case of a patient with advanced GCT arising in the spine, recurring after multiple resections and embolization. Following initiation of denosumab, which resulted in unequivocal clinical improvement, computed tomography of the chest done for reassessment purposes revealed an intratumoral pseudoaneurysm by erosion of the aorta, further corrected by endovascular approach and stent placement. Patient had an unremarkable recovery from the procedure and continued benefit from therapy with denosumab and remains on treatment 24 months after the first dose.
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institution Kabale University
issn 2090-6706
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language English
publishDate 2015-01-01
publisher Wiley
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series Case Reports in Oncological Medicine
spelling doaj-art-387c0fc7b0a9440db4494680f19a31f32025-02-03T05:46:18ZengWileyCase Reports in Oncological Medicine2090-67062090-67142015-01-01201510.1155/2015/626741626741Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to DenosumabNatalia M. P. Fraile0Diego Toloi1Ceci O. Kurimori2Adriana R. B. Matutino3Alberto Codima4Veridiana P. Camargo5Olavo Feher6Rodrigo R. Munhoz7Instituto do Câncer do Estado de São Paulo, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilInstituto do Câncer do Estado de São Paulo, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilInstituto do Câncer do Estado de São Paulo, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilInstituto do Câncer do Estado de São Paulo, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilHospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilInstituto do Câncer do Estado de São Paulo, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilInstituto do Câncer do Estado de São Paulo, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilInstituto do Câncer do Estado de São Paulo, Faculdade de Medicina da Universidade de São Paulo, São Paulo, SP, BrazilGiant cell tumor of bone (GCT) is a rare, locally aggressive neoplasm characterized by the presence of giant cells with osteoclast activity. Its biology involves the overexpression of the Receptor Activator of Nuclear Factor kB Ligand (RANKL) by osteoclast-like giant cells and tumor stromal cells, which has been shown to be an actionable target in this disease. In cases amenable to surgical resection, very few therapeutic options were available until the recent demonstration of significant activity of the anti-RANK-ligand monoclonal antibody denosumab. Here we present a case of a patient with advanced GCT arising in the spine, recurring after multiple resections and embolization. Following initiation of denosumab, which resulted in unequivocal clinical improvement, computed tomography of the chest done for reassessment purposes revealed an intratumoral pseudoaneurysm by erosion of the aorta, further corrected by endovascular approach and stent placement. Patient had an unremarkable recovery from the procedure and continued benefit from therapy with denosumab and remains on treatment 24 months after the first dose.http://dx.doi.org/10.1155/2015/626741
spellingShingle Natalia M. P. Fraile
Diego Toloi
Ceci O. Kurimori
Adriana R. B. Matutino
Alberto Codima
Veridiana P. Camargo
Olavo Feher
Rodrigo R. Munhoz
Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to Denosumab
Case Reports in Oncological Medicine
title Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to Denosumab
title_full Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to Denosumab
title_fullStr Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to Denosumab
title_full_unstemmed Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to Denosumab
title_short Successful Intravascular Correction of Intratumoral Pseudoaneurysm by Erosion of the Aorta in a Patient with Thoracic Giant Cell Tumor of Bone Responding to Denosumab
title_sort successful intravascular correction of intratumoral pseudoaneurysm by erosion of the aorta in a patient with thoracic giant cell tumor of bone responding to denosumab
url http://dx.doi.org/10.1155/2015/626741
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