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Published in: Skeletal Radiology 4/2017

01-04-2017 | Case Report

Giant cell tumor of the bone: aggressive case initially treated with denosumab and intralesional surgery

Authors: Donald von Borstel, D.O., Roberto A. Taguibao, M.D., Nicholas A. Strle, D.O., Joseph E. Burns, M.D., Ph.D.

Published in: Skeletal Radiology | Issue 4/2017

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Abstract

Giant cell tumor of the bone (GCTB) is a locally aggressive benign tumor, which has historically been treated with wide surgical excision. We report a case of a 29-year-old male with histology-proven GCTB of the distal ulna. The initial imaging study was a contrast-enhanced magnetic resonance imaging (MRI) examination of the left wrist, which was from an outside facility performed before presenting to our institution. On the initial MRI, the lesion had homogenous T2-hyperintense and T1-hypointense signal with expansive remodeling of the osseous contour. A radiographic study performed upon presentation to our institution 1 month later showed progression of the lesion with atypical imaging characteristics. After confirming the diagnosis, denosumab therapy was implemented allowing for reconstitution of bone and intralesional treatment. The patient was treated with five doses of denosumab over the duration of 7 weeks. Therapeutic changes of the GCTB were evaluated by radiography and a post-treatment MRI. This MRI was interpreted as suspicious for worsening disease due to the imaging appearance of intralesional signal heterogeneity, increased perilesional fluid-like signal, and circumferential cortical irregularity. However, on subsequent intralesional curettage and bone autografting 6 weeks later, no giant cells were seen on the specimen. Thus, the appearance on the MRI, rather than representing a manifestation of lesion aggressiveness or a non-responding tumor, conversely represented the imaging appearance of a positive response to denosumab therapy. On follow-up evaluation, 5 months after intralesional treatment, the patient had recurrent disease and is now scheduled for wide-excision with joint prosthesis.
Literature
1.
go back to reference Murphey M, Nomikos G, Flemming D, Gannon F, Temple T, Kransdorf M. Imaging of giant cell tumor and giant cell reparative granuloma of bone: radiologic-pathologic correlation. Radiographics. 2001;21(5):1203–309.CrossRef Murphey M, Nomikos G, Flemming D, Gannon F, Temple T, Kransdorf M. Imaging of giant cell tumor and giant cell reparative granuloma of bone: radiologic-pathologic correlation. Radiographics. 2001;21(5):1203–309.CrossRef
2.
go back to reference Kalil RK. Giant cell tumor of bone. In: Santini-Araujo E, Kalil RK, Bertoni F, Park Y-K, editors. Tumors and tumor-like lesions of bone. London: Springer; 2015. p. 351–66.CrossRef Kalil RK. Giant cell tumor of bone. In: Santini-Araujo E, Kalil RK, Bertoni F, Park Y-K, editors. Tumors and tumor-like lesions of bone. London: Springer; 2015. p. 351–66.CrossRef
3.
go back to reference Chakarun C, Forrester D, Gottsegen C, Patel D, White E, Matcuk G. Giant cell tumor of bone: review, mimics, and new developments in treatment. Radiographics. 2013;33(1):197–211.CrossRefPubMed Chakarun C, Forrester D, Gottsegen C, Patel D, White E, Matcuk G. Giant cell tumor of bone: review, mimics, and new developments in treatment. Radiographics. 2013;33(1):197–211.CrossRefPubMed
5.
go back to reference Roux S, Amazit L, Meduri G, Guiochon-Mantel A, Milgrom E, Mariette X. RANK (receptor activotor of nuclear factor kappa B) and RANK ligand are expressed in giant cell tumors of bone. Am J Clin Pathol. 2002;117:210–6.CrossRefPubMed Roux S, Amazit L, Meduri G, Guiochon-Mantel A, Milgrom E, Mariette X. RANK (receptor activotor of nuclear factor kappa B) and RANK ligand are expressed in giant cell tumors of bone. Am J Clin Pathol. 2002;117:210–6.CrossRefPubMed
6.
go back to reference Chawla S, Henshaw R, Seeger L, Choy E, Blay J, Ferrari S. Safety and efficacy of denosumab for adults and skeletally mature adolescents with giant cell tumour of bone: interim analysis of an open-label, parallel-group, phase 2 study. Lancet Oncol. 2013;14(9):901–8.CrossRefPubMed Chawla S, Henshaw R, Seeger L, Choy E, Blay J, Ferrari S. Safety and efficacy of denosumab for adults and skeletally mature adolescents with giant cell tumour of bone: interim analysis of an open-label, parallel-group, phase 2 study. Lancet Oncol. 2013;14(9):901–8.CrossRefPubMed
7.
go back to reference Thomas D, Henshaw R, Skubitz K, Chawla S, Staddon A, Blay J-Y, et al. Denosumab in patients with giant-cell tumour of bone: an open-label, phase 2 study. Lancet Oncol. 2010;11(3):275–80.CrossRefPubMed Thomas D, Henshaw R, Skubitz K, Chawla S, Staddon A, Blay J-Y, et al. Denosumab in patients with giant-cell tumour of bone: an open-label, phase 2 study. Lancet Oncol. 2010;11(3):275–80.CrossRefPubMed
8.
go back to reference Hu MI, Glezerman I, Leboulleux S, Insogna K, Gucalp R, Misiorowski W, et al. Denosumab for patients with persistent or relapsed hypercalcemia of malignancy despite recent bisphosphonate treatment. J Natl Cancer Inst. 2013;105(18):1417–20.CrossRefPubMedPubMedCentral Hu MI, Glezerman I, Leboulleux S, Insogna K, Gucalp R, Misiorowski W, et al. Denosumab for patients with persistent or relapsed hypercalcemia of malignancy despite recent bisphosphonate treatment. J Natl Cancer Inst. 2013;105(18):1417–20.CrossRefPubMedPubMedCentral
9.
go back to reference Lipton A, Fizazi K, Stopeck AT, Henry DH, Brown JE, Yardley DA, et al. Superiority of denosumab to zoledronic acid for prevention of skeletal-related events: A combined analysis of 3 pivotal, randomised, phase 3 trials. Eur J Cancer. 2012;48(16):3082–92.CrossRefPubMed Lipton A, Fizazi K, Stopeck AT, Henry DH, Brown JE, Yardley DA, et al. Superiority of denosumab to zoledronic acid for prevention of skeletal-related events: A combined analysis of 3 pivotal, randomised, phase 3 trials. Eur J Cancer. 2012;48(16):3082–92.CrossRefPubMed
10.
go back to reference Gerdes J, Schwab U, Lemke H, Stein H. Production of a mouse monoclonal antibody reactive with a human nuclear antigen associated with cell proliferation. Int J Cancer. 1983;31(1):13–20.CrossRefPubMed Gerdes J, Schwab U, Lemke H, Stein H. Production of a mouse monoclonal antibody reactive with a human nuclear antigen associated with cell proliferation. Int J Cancer. 1983;31(1):13–20.CrossRefPubMed
11.
go back to reference Fletcher C, Bridge J, Hogendoorn P, Mertens F. WHO classification of tumour of soft tissue and bone. 4th ed. Lyon: IARC Press; 2013. Fletcher C, Bridge J, Hogendoorn P, Mertens F. WHO classification of tumour of soft tissue and bone. 4th ed. Lyon: IARC Press; 2013.
12.
go back to reference Branstetter D, Nelson S, Manivel JC, Blay J-Y, Chawla S, Thomas D, et al. Denosumab induces tumor reduction and bone formation in patients with giant-cell tumor of bone. Clin Cancer Res. 2012;18(16):4415–24.CrossRefPubMed Branstetter D, Nelson S, Manivel JC, Blay J-Y, Chawla S, Thomas D, et al. Denosumab induces tumor reduction and bone formation in patients with giant-cell tumor of bone. Clin Cancer Res. 2012;18(16):4415–24.CrossRefPubMed
14.
go back to reference Ueda T, Morioka H, Nishida Y, Kakunaga S, Tsuchiya H, Matsumoto Y, et al. Objective tumor response to denosumab in patients with giant cell tumor of bone: a multicenter phase II trial. Ann Oncol. 2015;26(10):2149–54. Ueda T, Morioka H, Nishida Y, Kakunaga S, Tsuchiya H, Matsumoto Y, et al. Objective tumor response to denosumab in patients with giant cell tumor of bone: a multicenter phase II trial. Ann Oncol. 2015;26(10):2149–54.
15.
go back to reference Campanacci M, Baldini N, Boriani S, Sudanesi A. Giant cell tumor of bone. J Bone Joint Surg. 1987;69(1):106–14.CrossRefPubMed Campanacci M, Baldini N, Boriani S, Sudanesi A. Giant cell tumor of bone. J Bone Joint Surg. 1987;69(1):106–14.CrossRefPubMed
16.
go back to reference Wang H-C, Chien S-H. G-T. L. Management of grade III giant cell tumors of bones. J Surg Oncol. 2005;18(16):4415–24. Wang H-C, Chien S-H. G-T. L. Management of grade III giant cell tumors of bones. J Surg Oncol. 2005;18(16):4415–24.
17.
go back to reference Rutkowski P, Ferrari S, Grimer R, Stalley P, Dijkstra S, Pienkowski A, et al. Surgical downstaging in an open-label phase ii trial of denosumab in patients with giant cell tumor of bone. Ann Surg Oncol. 2015;22(9):2860–8.CrossRefPubMedPubMedCentral Rutkowski P, Ferrari S, Grimer R, Stalley P, Dijkstra S, Pienkowski A, et al. Surgical downstaging in an open-label phase ii trial of denosumab in patients with giant cell tumor of bone. Ann Surg Oncol. 2015;22(9):2860–8.CrossRefPubMedPubMedCentral
18.
go back to reference Klenke F, Wenger D, Inwards C, Rose P, Sim F. Recurrent giant cell tumor of long bones: analysis of surgical management. Clin Orthop Relat Res. 2011;469(4):1181–7.CrossRefPubMed Klenke F, Wenger D, Inwards C, Rose P, Sim F. Recurrent giant cell tumor of long bones: analysis of surgical management. Clin Orthop Relat Res. 2011;469(4):1181–7.CrossRefPubMed
19.
go back to reference Jagiello-Wieczorek E, Pienkowski A, Rutkowski P. Denosumab for treating giant cell tumor of bone. Expert Opin Orphan Drugs. 2015;3(10):1219–29.CrossRef Jagiello-Wieczorek E, Pienkowski A, Rutkowski P. Denosumab for treating giant cell tumor of bone. Expert Opin Orphan Drugs. 2015;3(10):1219–29.CrossRef
Metadata
Title
Giant cell tumor of the bone: aggressive case initially treated with denosumab and intralesional surgery
Authors
Donald von Borstel, D.O.
Roberto A. Taguibao, M.D.
Nicholas A. Strle, D.O.
Joseph E. Burns, M.D., Ph.D.
Publication date
01-04-2017
Publisher
Springer Berlin Heidelberg
Published in
Skeletal Radiology / Issue 4/2017
Print ISSN: 0364-2348
Electronic ISSN: 1432-2161
DOI
https://doi.org/10.1007/s00256-017-2588-7

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