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BMC Cancer
Published in: BMC Cancer 1/2007

Open Access 01-12-2007 | Case report

Giant cell tumor of the uterus: case report and response to chemotherapy

Authors: Keith M Skubitz, Js Carlos Manivel

Published in: BMC Cancer | Issue 1/2007

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Abstract

Background

Giant cell tumor (GCT) is usually a benign but locally aggressive primary bone neoplasm in which monocytic macrophage/osteoclast precursor cells and multinucleated osteoclast-like giant cells infiltrate the tumor. The etiology of GCT is unknown, however the tumor cells of GCT have been reported to produce chemoattractants that can attract osteoclasts and osteoclast precursors. Rarely, GCT can originate at extraosseous sites. More rarely, GCT may exhibit a much more aggressive phenotype. The role of chemotherapy in metastatic GCT is not well defined.

Case presentation

We report a case of an aggressive GCT of the uterus with rapidly growing lung metastases, and its response to chemotherapy with pegylated-liposomal doxorubicin, ifosfamide, and bevacizumab, along with a review of the literature.

Conclusion

Aggressive metastasizing GCT may arise in the uterus, and may respond to combination chemotherapy.
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Literature
1.
Athanasou NA, Bliss E, Gatter KC, Heryet A, Woods CG, McGee JO: An immunohistological study of giant-cell tumour of bone: evidence for an osteoclast origin of the giant cells. J Pathol. 1985, 147: 153-158. 10.1002/path.1711470302.CrossRefPubMed
2.
Goldring SR, Roelke MS, Petrison KK, Bhan AK: Human giant cell tumors of bone identification and characterization of cell types. J Clin Invest. 1987, 79: 483-491.CrossRefPubMedPubMedCentral
3.
Zheng MH, Fan Y, Smith A, Wysocki S, Papadimitriou JM, Wood DJ: Gene expression of monocyte chemoattractant protein-1 in giant cell tumors of bone osteoclastoma: possible involvement in CD68+ macrophage-like cell migration. J Cell Biochem. 1998, 70: 121-129. 10.1002/(SICI)1097-4644(19980701)70:1<121::AID-JCB12>3.0.CO;2-S.CrossRefPubMed
4.
Walker DG: Congenital osteopetrosis in mice cured by parabiotic union with normal siblings. Endocrinology. 1972, 91: 916-920.CrossRefPubMed
5.
Walker DG: Bone resorption restored in osteopetrotic mice by transplants of normal bone marrow and spleen cells. Science. 1975, 190: 784-785. 10.1126/science.1105786.CrossRefPubMed
6.
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: 210-216. 10.1309/BPET-F2PE-P2BD-J3P3.CrossRefPubMed
7.
Tubbs WS, Brown LR, Beabout JW, Rock MG, Unni KK: Benign giant-cell tumor of bone with pulmonary metastases: clinical findings and radiologic appearance of metastases in 13 cases. AJR Am J Roentgenol. 1992, 158: 331-334.CrossRefPubMed
8.
Skubitz KM, Hamdan H, Thompson RC: Ambulatory continuous infusion ifosfamide with oral etoposide in advanced sarcomas. Cancer. 1993, 72: 2963-2969. 10.1002/1097-0142(19931115)72:10<2963::AID-CNCR2820721017>3.0.CO;2-W.CrossRefPubMed
9.
Okuno S, Edmonson J, Mahoney M, Buckner JC, Frytak S, Galanis E: Phase II trial of gemcitabine in advanced sarcomas. Cancer. 2002, 94: 3225-3229. 10.1002/cncr.10602.CrossRefPubMed
10.
Patel SR, Gandhi V, Jenkins J, Papadopolous N, Burgess MA, Plager C, Plunkett W, Benjamin RS: Phase II clinical investigation of gemcitabine in advanced soft tissue sarcomas and window evaluation of dose rate on gemcitabine triphosphate accumulation. J Clin Oncol. 2001, 19: 3483-3489.PubMed
11.
Northfelt DW, Dezube BJ, Thommes JA, Miller BJ, Fischl MA, Friedman-Kien A, Kaplan LD, Du Mond C, Mamelok RD, Henry DH: Pegylated-liposomal doxorubicin versus doxorubicin, bleomycin, and vincristine in the treatment of AIDS-related Kaposi's sarcoma: results of a randomized phase III clinical trial. J Clin Oncol. 1998, 16: 2445-2451.PubMed
12.
Skubitz KM: Phase II trial of pegylated-liposomal doxorubicin (Doxil) in sarcoma. Cancer Invest. 2003, 21: 167-176. 10.1081/CNV-120016412.CrossRefPubMed
13.
Skubitz KM, Haddad PA: Paclitaxel and pegylated-liposomal doxorubicin are both active in angiosarcoma. Cancer. 2005, 104: 361-366. 10.1002/cncr.21140.CrossRefPubMed
14.
Judson I, Radford JA, Harris M, Blay JY, van Hoesel Q, le Cesne A, van Oosterom AT, Clemons MJ, Kamby C, Hermans C, Whittaker J, Donato di Paola E, Verweij J, Nielsen S: Randomised phase II trial of pegylated liposomal doxorubicin (DOXIL/CAELYX) versus doxorubicin in the treatment of advanced or metastatic soft tissue sarcoma: a study by the EORTC Soft Tissue and Bone Sarcoma Group. Eur J Cancer. 2001, 37: 870-877. 10.1016/S0959-8049(01)00050-8.CrossRefPubMed
15.
Patel SR, Vadhan-Raj S, Papadopolous N, Plager C, Burgess MA, Hays C, Benjamin RS: High-dose ifosfamide in bone and soft tissue sarcomas: results of phase II and pilot studies--dose-response and schedule dependence. J Clin Oncol. 1997, 15: 2378-2384.PubMed
16.
Zheng MH, Xu J, Robbins P, Pavlos N, Wysocki S, Kumta SM, Wood DJ, Papadimitriou JM: Gene expression of vascular endothelial growth factor in giant cell tumors of bone. Hum Pathol. 2000, 31: 804-812. 10.1053/hupa.2000.8441.CrossRefPubMed
17.
Skubitz KM, Skubitz AP: Characterization of sarcomas by means of gene expression. J Lab Clin Med. 2004, 144: 78-91. 10.1016/j.lab.2004.04.005.CrossRefPubMed
18.
Skubitz KM, Cheng EY, Clohisy DR, Thompson RC, Skubitz AP: Gene expression in giant-cell tumors. J Lab Clin Med. 2004, 144: 193-200. 10.1016/j.lab.2004.06.005.CrossRefPubMed
19.
Kumta SM, Huang L, Cheng YY, Chow LT, Lee KM, Zheng MH: Expression of VEGF and MMP-9 in giant cell tumor of bone and other osteolytic lesions. Life Sci. 2003, 73: 1427-1436. 10.1016/S0024-3205(03)00434-X.CrossRefPubMed
20.
Hoang BH, Dyke JP, Koutcher JA, Huvos AG, Mizobuchi H, Mazza BA, Gorlick R, Healey JH: VEGF expression in osteosarcoma correlates with vascular permeability by dynamic MRI. Clin Orthop Relat Res. 2004, 32-38. 10.1097/01.blo.0000141492.52166.20.
21.
Heymach JV, Desai J, Manola J, Davis DW, McConkey DJ, Harmon D, Ryan DP, Goss G, Quigley T, Van den Abbeele AD, Silverman SG, Connors S, Folkman J, Fletcher CD, Demetri GD: Phase II study of the antiangiogenic agent SU5416 in patients with advanced soft tissue sarcomas. Clin Cancer Res. 2004, 10: 5732-5740. 10.1158/1078-0432.CCR-04-0157.CrossRefPubMed
22.
Hayes AJ, Mostyn-Jones A, Koban MU, A'Hern R, Burton P, Thomas JM: Serum vascular endothelial growth factor as a tumour marker in soft tissue sarcoma. Br J Surg. 2004, 91: 242-247. 10.1002/bjs.4398.CrossRefPubMed
23.
Graeven U, Andre N, Achilles E, Zornig C, Schmiegel W: Serum levels of vascular endothelial growth factor and basic fibroblast growth factor in patients with soft-tissue sarcoma. J Cancer Res Clin Oncol. 1999, 125: 577-581. 10.1007/s004320050319.CrossRefPubMed
24.
Niida S, Kaku M, Amano H, Yoshida H, Kataoka H, Nishikawa S, Tanne K, Maeda N, Kodama H: Vascular endothelial growth factor can substitute for macrophage colony-stimulating factor in the support of osteoclastic bone resorption. J Exp Med. 1999, 190: 293-298. 10.1084/jem.190.2.293.CrossRefPubMedPubMedCentral
25.
van Haelst UJ, de Haas van Dorsser AH: Giant cell tumor of soft parts. An ultrastructural study. Virchows Arch A Pathol Anat Histol. 1976, 371: 199-217. 10.1007/BF00433068.CrossRefPubMed
26.
Dodd LG, Major N, Brigman B: Malignant giant cell tumor of soft parts. Skeletal Radiol. 2004, 33: 295-299. 10.1007/s00256-003-0744-8.CrossRefPubMed
27.
Silverberg SG, DeGiorgi LS: Osteoclastoma-like giant cell tumor of the thyroid. Report of a case with prolonged survival following partial excision and radiotherapy. Cancer. 1973, 31: 621-625. 10.1002/1097-0142(197303)31:3<621::AID-CNCR2820310319>3.0.CO;2-N.CrossRefPubMed
28.
Rosai J: Carcinoma of pancreas simulating giant cell tumor of bone. Electron-microscopic evidence of its acinar cell origin. Cancer. 1968, 22: 333-344. 10.1002/1097-0142(196808)22:2<333::AID-CNCR2820220210>3.0.CO;2-A.CrossRefPubMed
29.
Trepeta RW, Mathur B, Lagin S, LiVolsi VA: Giant cell tumor ("osteoclastoma") of the pancreas: a tumor of epithelial origin. Cancer. 1981, 48: 2022-2028. 10.1002/1097-0142(19811101)48:9<2022::AID-CNCR2820480919>3.0.CO;2-I.CrossRefPubMed
30.
Caudell JJ, Ballo MT, Zagars GK, Lewis VO, Weber KL, Lin PP, Marco RA, El-Naggar AK, Benjamin RS, Yasko AW: Radiotherapy in the management of giant cell tumor of bone. Int J Radiat Oncol Biol Phys. 2003, 57: 158-165. 10.1016/S0360-3016(03)00416-4.CrossRefPubMed
31.
Feigenberg SJ, Marcus RB, Zlotecki RA, Scarborough MT, Enneking WF: Whole-lung radiotherapy for giant cell tumors of bone with pulmonary metastases. Clin Orthop Relat Res. 2002, 202-208. 10.1097/00003086-200208000-00023.
32.
Osaka S, Toriyama M, Taira K, Sano S, Saotome K: Analysis of giant cell tumor of bone with pulmonary metastases. Clin Orthop Relat Res. 1997, 253-261.
33.
Maloney WJ, Vaughan LM, Jones HH, Ross J, Nagel DA: Benign metastasizing giant-cell tumor of bone. Report of three cases and review of the literature. Clin Orthop Relat Res. 1989, 208-215.
34.
Bertoni F, Present D, Sudanese A, Baldini N, Bacchini P, Campanacci M: Giant-cell tumor of bone with pulmonary metastases. Six case reports and a review of the literature. Clin Orthop Relat Res. 1988, 275-285.
35.
Bertoni F, Present D, Enneking WF: Giant-cell tumor of bone with pulmonary metastases. J Bone Joint Surg Am. 1985, 67: 890-900.PubMed
36.
Sanjay BK, Kadhi SM: Giant cell tumour of bone with pulmonary metastases. A report of three cases. Int Orthop. 1998, 22: 200-204. 10.1007/s002640050242.CrossRefPubMedPubMedCentral
37.
Stewart DJ, Belanger R, Benjamin RS: Prolonged disease-free survival following surgical debulking and high-dose cisplatin/doxorubicin in a patient with bulky metastases from giant cell tumor of bone refractory to "standard" chemotherapy. Am J Clin Oncol. 1995, 18: 144-148.CrossRefPubMed
38.
Yamamoto M, Fukushima T, Sakamoto S, Tomonaga M: Giant cell tumor of the sphenoid bone: long-term follow-up of two cases after chemotherapy. Surg Neurol. 1998, 49: 547-552. 10.1016/S0090-3019(97)00219-X.CrossRefPubMed
39.
Kay RM, Eckardt JJ, Seeger LL, Mirra JM, Hak DJ: Pulmonary metastasis of benign giant cell tumor of bone. Six histologically confirmed cases, including one of spontaneous regression. Clin Orthop Relat Res. 1994, 219-230.
40.
Lachat MR, Weber M, Cserhati MD, Honegger HP, von Hochstetter AR: [Giant cell tumor of bone with rapid malignant course]. Orthopade. 2004, 33: 344-348. 10.1007/s00132-003-0554-7.CrossRefPubMed
41.
De Smedt M, Copin G, Boeri C, Dosch JC, Dupuis M, Marcellin L: [A case of a aggressive giant-cell tumor with multiple bone metastases]. Rev Chir Orthop Reparatrice Appar Mot. 1999, 85: 293-296.PubMed
42.
Kaban LB, Troulis MJ, Ebb D, August M, Hornicek FJ, Dodson TB: Antiangiogenic therapy with interferon alpha for giant cell lesions of the jaws. J Oral Maxillofac Surg. 2002, 60: 1103-11; discussion 1111-3. 10.1053/joms.2002.34975.CrossRefPubMed
43.
Kaiser U, Neumann K, Havemann K: Generalised giant-cell tumour of bone: successful treatment of pulmonary metastases with interferon alpha, a case report. J Cancer Res Clin Oncol. 1993, 119: 301-303. 10.1007/BF01212729.CrossRefPubMed
44.
Dickerman JD: Interferon and giant cell tumors. Pediatrics. 1999, 103: 1282-1283. 10.1542/peds.103.6.1282.CrossRefPubMed
45.
Magni E, Lauritzen AF, Wilken-Jensen C, Horn T: Malignant giant cell tumour of the uterus. APMIS Suppl. 1991, 23: 113-118.PubMed
46.
Sieinski W: Malignant giant cell tumor associated with leiomyosarcoma of the uterus. Cancer. 1990, 65: 1838-1842. 10.1002/1097-0142(19900415)65:8<1838::AID-CNCR2820650829>3.0.CO;2-1.CrossRefPubMed
47.
Watanabe K, Hiraki H, Ohishi M, Mashiko K, Saginoya H, Suzuki T: Uterine leiomyosarcoma with osteoclast-like giant cells: histopathological and cytological observations. Pathol Int. 1996, 46: 656-660.CrossRefPubMed
48.
Egorov VP, Naumov VN: [Malignant giant cell uterine tumors]. Arkh Patol. 1983, 45: 78-82.PubMed
49.
Kindblom LG, Seidal T: Malignant giant cell tumor of the uterus. A clinico-pathologic, light- and electron-microscopic study of a case. Acta Pathol Microbiol Scand [A]. 1981, 89: 179-184.
50.
Darby AJ, Papadaki L, Beilby JO: An unusual leiomyosarcoma of the uterus containing osteoclast-like giant cells. Cancer. 1975, 36: 495-504. 10.1002/1097-0142(197508)36:2<495::AID-CNCR2820360228>3.0.CO;2-I.CrossRefPubMed
51.
Pilon VA, Parikh N, Maccera J: Malignant osteoclast-like giant cell tumor associated with a uterine leiomyosarcoma. Gynecol Oncol. 1986, 23: 381-386. 10.1016/0090-8258(86)90142-3.CrossRefPubMed
52.
Huang L, Xu J, Wood DJ, Zheng MH: Gene expression of osteoprotegerin ligand, osteoprotegerin, and receptor activator of NF-kappaB in giant cell tumor of bone: possible involvement in tumor cell-induced osteoclast-like cell formation. Am J Pathol. 2000, 156: 761-767.CrossRefPubMedPubMedCentral
53.
Kong YY, Feige U, Sarosi I, Bolon B, Tafuri A, Morony S, Capparelli C, Li J, Elliott R, McCabe S, Wong T, Campagnuolo G, Moran E, Bogoch ER, Van G, Nguyen LT, Ohashi PS, Lacey DL, Fish E, Boyle WJ, Penninger JM: Activated T cells regulate bone loss and joint destruction in adjuvant arthritis through osteoprotegerin ligand. Nature. 1999, 402: 304-309. 10.1038/46303.CrossRefPubMed
54.
Atkins GJ, Haynes DR, Graves SE, Evdokiou A, Hay S, Bouralexis S, Findlay DM: Expression of osteoclast differentiation signals by stromal elements of giant cell tumors. J Bone Miner Res. 2000, 15: 640-649. 10.1359/jbmr.2000.15.4.640.CrossRefPubMed
55.
Lacey DL, Timms E, Tan HL, Kelley MJ, Dunstan CR, Burgess T, Elliott R, Colombero A, Elliott G, Scully S, Hsu H, Sullivan J, Hawkins N, Davy E, Capparelli C, Eli A, Qian YX, Kaufman S, Sarosi I, Shalhoub V, Senaldi G, Guo J, Delaney J, Boyle WJ: Osteoprotegerin ligand is a cytokine that regulates osteoclast differentiation and activation. Cell. 1998, 93: 165-176. 10.1016/S0092-8674(00)81569-X.CrossRefPubMed
56.
Chang SS, Suratwala SJ, Jung KM, Doppelt JD, Zhang HZ, Blaine TA, Kim TW, Winchester RJ, Lee FY: Bisphosphonates may reduce recurrence in giant cell tumor by inducing apoptosis. Clin Orthop Relat Res. 2004, 103-109. 10.1097/01.blo.0000141372.54456.80.
57.
Cheng YY, Huang L, Lee KM, Xu JK, Zheng MH, Kumta SM: Bisphosphonates induce apoptosis of stromal tumor cells in giant cell tumor of bone. Calcif Tissue Int. 2004, 75: 71-77. 10.1007/s00223-004-0120-2.CrossRefPubMed
Metadata
Title
Giant cell tumor of the uterus: case report and response to chemotherapy
Authors
Keith M Skubitz
Js Carlos Manivel
Publication date
01-12-2007
Publisher
BioMed Central
Published in
BMC Cancer / Issue 1/2007
Electronic ISSN: 1471-2407
DOI
https://doi.org/10.1186/1471-2407-7-46

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