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Published in: Skeletal Radiology 12/2014

01-12-2014 | Case Report

An eggshell-like mineralized recurrent lesion in the popliteal region after treatment of giant cell tumor of the bone with denosumab

Authors: Keisuke Akaike, Yoshiyuki Suehara, Tatsuya Takagi, Kazuo Kaneko, Tsuyoshi Saito

Published in: Skeletal Radiology | Issue 12/2014

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Abstract

We report a case of recurrent giant cell tumor of the bone (GCTB) in which treatment with denosumab gradually enhanced the eggshell-like mineralization at the periphery of the tumor. A 28-year-old male presented with a mass on his left distal femur that had enlarged within the past few months. Before curettage, GCTB of the distal femur was diagnosed based on histological analysis of a biopsy specimen; the tumor consisted of a proliferation of ovoid mononuclear stromal cells with evenly scattered multinucleated osteoclast-like giant cells. The tumor recurred three times after the initial diagnosis; at the time of the third relapse, the patient underwent en bloc resection and reconstruction with a knee joint prosthesis. He was also treated with denosumab postoperatively because some studies have recently shown the benefits of the receptor activator of nuclear factor kappa-B ligand (RANKL) inhibitor denosumab as adjuvant therapy in patients with GCTB. Six months after starting adjuvant treatment with denosumab, radiography revealed a mineralized nodule >2 cm in diameter at the popliteal region; this lesion was considered a soft tissue recurrence of GCTB. Treatment with denosumab was continued for another 1.5 years, and the lesion was resected. Histological examination showed residual mononuclear stromal cells expressing RANKL without multinucleated giant cells surrounded by the peripheral mineralization. The patient was successfully treated by complete resection with the support of adjuvant treatment with denosumab.
Literature
1.
go back to reference Cooper KL, Beabout JW, Dahlin DC. Giant cell tumor: ossification in soft tissue implants. Radiology. 1984;153:597–602.PubMedCrossRef Cooper KL, Beabout JW, Dahlin DC. Giant cell tumor: ossification in soft tissue implants. Radiology. 1984;153:597–602.PubMedCrossRef
2.
go back to reference Ehara S, Nishida J, Abe M, Kawata Y, Saitoh H, Kattapuram SV. Ossified soft tissue recurrence of giant cell tumor of bone. Clin Imaging. 1992;16:168–71.PubMedCrossRef Ehara S, Nishida J, Abe M, Kawata Y, Saitoh H, Kattapuram SV. Ossified soft tissue recurrence of giant cell tumor of bone. Clin Imaging. 1992;16:168–71.PubMedCrossRef
3.
go back to reference Lee FY, Montgomery M, Hazan EJ, Keel SB, Mankin HJ, Kattapuram S. Recurrent giant-cell tumor presenting as a soft-tissue mass. A report of four cases. J Bone Joint Surg Am. 1999;81:703–7.PubMedCrossRef Lee FY, Montgomery M, Hazan EJ, Keel SB, Mankin HJ, Kattapuram S. Recurrent giant-cell tumor presenting as a soft-tissue mass. A report of four cases. J Bone Joint Surg Am. 1999;81:703–7.PubMedCrossRef
4.
go back to reference Oliveira AM, Dei Tos AP, Fletcher CD, Nascimento AG. Primary giant cell tumor of soft tissues: a study of 22 cases. Am J Surg Pathol. 2000;24:248–56.PubMedCrossRef Oliveira AM, Dei Tos AP, Fletcher CD, Nascimento AG. Primary giant cell tumor of soft tissues: a study of 22 cases. Am J Surg Pathol. 2000;24:248–56.PubMedCrossRef
5.
go back to reference O’Connell JX, Wehrli BM, Nielsen GP, Rosenberg AE. Giant cell tumors of soft tissue: a clinicopathologic study of 18 benign and malignant tumors. Am J Surg Pathol. 2000;24:386–95.PubMedCrossRef O’Connell JX, Wehrli BM, Nielsen GP, Rosenberg AE. Giant cell tumors of soft tissue: a clinicopathologic study of 18 benign and malignant tumors. Am J Surg Pathol. 2000;24:386–95.PubMedCrossRef
6.
go back to reference Roux S, Amazit L, Meduri G, Guiochon-Mantel A, Milgrom E, Mariette X. RANK (receptor activator of nuclear factor kappa B) and RANK ligand are expressed in giant cell tumors of bone. Am J Clin Pathol. 2002;117:210–6.PubMedCrossRef Roux S, Amazit L, Meduri G, Guiochon-Mantel A, Milgrom E, Mariette X. RANK (receptor activator of nuclear factor kappa B) and RANK ligand are expressed in giant cell tumors of bone. Am J Clin Pathol. 2002;117:210–6.PubMedCrossRef
7.
go back to reference Thomas D, Henshaw R, Skubitz K, Chawla S, Staddon A, Blay JY, etal. Denosumab in patients with giant-cell tumour of bone: an open-label, phase 2 study. Lancet Oncol. 2010;11:275–80. Thomas D, Henshaw R, Skubitz K, Chawla S, Staddon A, Blay JY, etal. Denosumab in patients with giant-cell tumour of bone: an open-label, phase 2 study. Lancet Oncol. 2010;11:275–80.
8.
go back to reference Branstetter DG, Nelson SD, Manivel JC, Blay JK, Chawla S, Thomas DM, etal. Denosumab induces tumor reduction and bone formation in patients with giant-cell tumor of bone. Clin Cancer Res. 2012;18:4415–24. Branstetter DG, Nelson SD, Manivel JC, Blay JK, Chawla S, Thomas DM, etal. Denosumab induces tumor reduction and bone formation in patients with giant-cell tumor of bone. Clin Cancer Res. 2012;18:4415–24.
9.
go back to reference Chawla S, Henshaw R, Seeger L, Choy E, Blay JY, Ferrari S, etal. 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;14:901–8. Chawla S, Henshaw R, Seeger L, Choy E, Blay JY, Ferrari S, etal. 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;14:901–8.
10.
go back to reference Okubo T, Saito T, Mitomi H, Takagi T, Torigoe T, Suehara Y, etal. P53 mutations may be involved in malignant transformation of giant cell tumor of bone through interaction with GPX1. Virchows Arch. 2013;463:67–77. Okubo T, Saito T, Mitomi H, Takagi T, Torigoe T, Suehara Y, etal. P53 mutations may be involved in malignant transformation of giant cell tumor of bone through interaction with GPX1. Virchows Arch. 2013;463:67–77.
11.
go back to reference Raskin KA, Schwab JH, Mankin HJ, Springfield DS, Homicek FJ. Giant cell tumor of bone. J Am Acad Orthop Surg. 2013;21:118–26.PubMedCrossRef Raskin KA, Schwab JH, Mankin HJ, Springfield DS, Homicek FJ. Giant cell tumor of bone. J Am Acad Orthop Surg. 2013;21:118–26.PubMedCrossRef
12.
go back to reference Hakozaki M, Tajino T, Yamada H, Hasegawa O, Tasaki K, Watanabe K, etal. Radiological and pathological characteristics of giant cell tumor of bone treated with denosumab. Diagn Pathol. 2014;9:111. Hakozaki M, Tajino T, Yamada H, Hasegawa O, Tasaki K, Watanabe K, etal. Radiological and pathological characteristics of giant cell tumor of bone treated with denosumab. Diagn Pathol. 2014;9:111.
13.
14.
go back to reference Teot LA, O’Keefe RJ, Rosier RN, O’Connell JX, Fox EJ, Hicks DG. Extraosseous primary and recurrent giant cell tumors: transforming growth factor-β1 and -β2 expression may explain metaplastic bone formation. Hum Pathol. 1996;27:625–32.PubMedCrossRef Teot LA, O’Keefe RJ, Rosier RN, O’Connell JX, Fox EJ, Hicks DG. Extraosseous primary and recurrent giant cell tumors: transforming growth factor-β1 and -β2 expression may explain metaplastic bone formation. Hum Pathol. 1996;27:625–32.PubMedCrossRef
15.
go back to reference Derbel O, Zrounba P, Chassagne-Clément C, Decouvelaere AV, Orlandini F, Duplomb S, etal. An unusual giant cell tumor of the thyroid: case report and review of the literature. J Clin Endocrinol Metab. 2013;98:1–6. Derbel O, Zrounba P, Chassagne-Clément C, Decouvelaere AV, Orlandini F, Duplomb S, etal. An unusual giant cell tumor of the thyroid: case report and review of the literature. J Clin Endocrinol Metab. 2013;98:1–6.
16.
go back to reference Agarwal A, Larsen BT, Buadu LD, Dunn J, Crawford R, Daniel J, etal. Denosumab chemotherapy for recurrent giant-cell tumor of bone: a case report of neoadjuvant use enabling complete surgical resection. Case Rep Oncol Med 2013, 496351. Agarwal A, Larsen BT, Buadu LD, Dunn J, Crawford R, Daniel J, etal. Denosumab chemotherapy for recurrent giant-cell tumor of bone: a case report of neoadjuvant use enabling complete surgical resection. Case Rep Oncol Med 2013, 496351.
17.
go back to reference Karras NA, Polgreen LE, Ogilvie C, Manivel JC, Skubitz KM, Lipsitz E. Denosumab treatment of metastatic giant-cell tumor of bone in a 10-year-old girl. J Clin Oncol. 2013;31:e200–2.PubMedCrossRef Karras NA, Polgreen LE, Ogilvie C, Manivel JC, Skubitz KM, Lipsitz E. Denosumab treatment of metastatic giant-cell tumor of bone in a 10-year-old girl. J Clin Oncol. 2013;31:e200–2.PubMedCrossRef
18.
go back to reference Toda M, Suehara Y, Akaike K, Okubo T, Kubota D, Mukaihara K, etal. Giant-cell tumor of bone arising in the anterior rib with a positive expression of GPX-1: a case report and review of the literature. Int Canc Conf J. 2013. doi:10.1007/s13691-013-0142-6. Toda M, Suehara Y, Akaike K, Okubo T, Kubota D, Mukaihara K, etal. Giant-cell tumor of bone arising in the anterior rib with a positive expression of GPX-1: a case report and review of the literature. Int Canc Conf J. 2013. doi:10.​1007/​s13691-013-0142-6.
Metadata
Title
An eggshell-like mineralized recurrent lesion in the popliteal region after treatment of giant cell tumor of the bone with denosumab
Authors
Keisuke Akaike
Yoshiyuki Suehara
Tatsuya Takagi
Kazuo Kaneko
Tsuyoshi Saito
Publication date
01-12-2014
Publisher
Springer Berlin Heidelberg
Published in
Skeletal Radiology / Issue 12/2014
Print ISSN: 0364-2348
Electronic ISSN: 1432-2161
DOI
https://doi.org/10.1007/s00256-014-1961-z

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