Giant Cell-Rich Osteosarcoma Simulating. Giant Cell Tumor of Bone

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1 Kitakanto Med J ; 54 : Giant Cell-Rich Osteosarcoma Simulatg Giant Cell Tum Bone Tetsuya Shozaki,1 Toshio Fukuda,2 Hideomi Watanabe,1 Kenji Takagishi1 A seventeen-year-old boy referred to our hospital, complag contuous pa his left wrist jot. Pla roentgenogram showed an osteolytic lesion at distal end radius. An operation which tum curetted perfmed suspectg cell tum. Pathological diagnosis a cell tum. One month after operation, radiological fdgs showed an exped lysis. Wide resection this tum followed by a vascularized fibular graft perfmed. Six months after second operation, st part swellg suggestg aga proment. Upper arm amputation perfmed bad condition. The pathological diagnosis se operative specimens cell tum similar to itial specimen. Multiple appeared eight months after third operation. In spite tensive treatment, patient died respiraty failure. Autopsy revealed pathological features metastatic specimens were similar to those osteosarcoma, not cell tum. When we encounter patients presentg a cell tum, especially when y are younger than age at which such lesions usually occur, we should bear md possibility an osteosarcoma perfm tensive chemapy surgery. (Kitakanto Med J 2004 ; 54 : 147 `151) Key wds : cell tum, osteosarcoma, autopsy, clical diagnosis, surgical treatment Introduction Giant cell tum bone comprises a characteristic mixture varyg numbers multucleated dispersed mononuclear.l2 Its pathological features are utilized to assess clical behavi.' prognostic significance histological gradg remas controversial.4 `6 Meover, aggressiveness on clical radiological grounds cannot be crelated any specific fdgs.4,6 Metastases are rare events : pulmonary have been repted less than 5 % patients conventional cell tum bone.5 `8 It is sometimes stressful f clicians to decide how to treat cell tum bone. Giant cell-rich osteosarcoma is a rare tum sub accountg f only 1-2 % all osteosarcomas.9,10 Increct histological diagnosis as a cell tum may be made similarities between se tums. Clical fmation cludg prognosis optical treatment is limited.10 This rept describes a case rare cell-rich osteosarcoma an aggressive clical course rapidly fatal outcome. Case Rept A seventeen-year-old boy referred to our hospital on August 1991 complag contuous pa his left wrist jot. He had itially noted pa May 1991 while playg volleyball. On physical examation, his wrist jot showed severe swellg heat tenderness ; range motion his left wrist jot slightly limited. Labaty data showed only a slightly elevated serum alkale phosphatase activity. Pla roentgenogram showed an osteolytic lesion at distal end radius (Fig. 1). Arteriography demonstrated hypervascularity at area. An operation by which tum 1 Department Orthopaedic Surgery, Gunma University Graduate School Medice 2 Department Labaty Sciences & Pathology, Gunma University School Health Sciences Received : December 12, 2003 Address : TETSUYA SHINOZAKI Department Orthopaedic Surgery, Gunma University Graduate School Medice, Showa Maebashi, Gunma, JAPAN

2 148 Giant Fig. 1 Pla roentgenogram presentation osteolytic left distal August radius The arrow Cell-Rich at time dicates lesion. Osteosarcomas Fig. 3 Pla roentgenogram showg exped lytic lesion loss sclerotic marg (arrow) September loss sclerotic marg (Fig. 3). Wide resection this tum followed by a vascularized fibular graft fusion left wrist jot perfmed October Pathological fdgs were similar to those at time itial operation. April 1992, st part swellg suggestg aga proment (Fig. 4). We consulted Fig. 2 Operative specimen, mitotic figures taken ovoid-shaped osteoclastic August 1991, showg a few (HE, ~ 50). curetted perfmed August Investigations lesions, such as bone extraskeletal, were not perfmed at time cell tum suspected clical radiological fdgs. Pathological diagnosis deed a cell tum ovoid-shaped, a few mitotic figures osteoclastic (Fig. 2). In September 1991, radiological fdgs his left wrist jot showed an exped lysis pathologists to confirm pathological fdgs were truly compatible those cell tum this unexpectedly early. They unanimously replied sefdgs were compatible those cell tum, not those osteosarcoma. Because deteriation clical fdgs, we started chemapy usg pirarubic, ifosfamide carboplat ; despite treatment, swellg wsened severe pa. Upper arm amputation perfmed September 1992 bad condition. Pathological fdgs were similar to those previous two operations. In December 1992, patient complaed low back pa. Pla roentgenography showed a compression fracture third lumbar vertebral body. Chest roentgenography also showed pulmonary metastasis at time. In June 1993, patient became unable to walk spal cd palsy below level

3 149 Fig. 5 Autopsy ary specimen, 1995, mitotic ous taken showg figures osteoclastic fmation right thax proment. Janua few Tum- (HE, ~ 75). pulmonary thacic wall (Fig. 5). They also showed a telangiectatic hemangiopericytoma-like pattern. Considerg se fdgs autopsy specimens, fal pathological diagnosis cell-rich osteosarcoma. Discussion Osteosarcomas are composed populations cludg cell-rich s similarity, benign st part swell- as anaplasia crect thacic spe. Gy, admistered In October creased acic 1993, 99mTc-DMSA uptake spe, Radiation rapy, to lumbar at skull, lumbar spe sctigraphy" sternum, spe, usg high-dose methotrexate, contued ; little total June cervical pelvis. cisplat effect showed spe, th- clical lesion curettage tion The case, couple ifosfamide wrist pulmonary lobes posteri thacic walls, thacic lumbar spes, sacral bone, lumbar spal cd, multiple monary walls tions. Little tumous posteri foci strifm operative fmation operative pattern specimens. proment specimens, pul- ossifica- multucleated atypism comprised a compact lung hemrhagic The mitoses. cluded terspersed showg Metastases a few differed diagnosis.9 to make presentg an fdg typical a after itial perfmed usg wide showed st aga. treatment Expectg Locally autograft. second swellg aggressive to reconstruc- fibular part appear a surgical resection after only alone.1,2 a vascularized months cell tum. noted be surgery, suggestg disease facts f risk pulmonary benign cell tum bone.7 Furr, primary lesion at distal creased cell tum sis compared ifosfamide, this radius risk lung lung dependent are imptant above, s especially several patient died January 1995 respiraty failure. At autopsy, were found bilateral upper skull. characteris- a defite radiological we tried roentgenography fdgs histological makg months control, differentiate features As we mentioned to Radiological diagnosis. osteolytic Chemapy obtaed. location can assist In our tenth GCT.9 tum such fmation Fig. 4 Pla roentgenogram showg proment g (arrow) July difficult malignant tics cell.12 Whereas repts are fewa) histological it is ten cludg heterogeneous on spontaneous we itiated aggressive seem to be associated.13 Patients an a had favable progno- used7,8,13 regression treatment chemapy carboplat radiological fdg repted.8,13,14 usg pirarubic, we suspected might show a

4 150 Giant Cell-Rich Osteosarcomas malignant bone tum such as osteosarcoma. when we consulted pathologists about pathological features operation specimens, y replied consistently features were characteristic a conventional cell tum, not osteosarcoma malignant cell tum. In spite tensive chemapy radiation rapy, patient died progressive. Autopsy revealed pathological features metastatic lesions showg tumous fmation were similar to those osteosarcoma, not cell tum. Malignant transfmation cell tum is less likely malignant transfmation fmerly conventional lesion should take many years out pri histy such as irradiation. It is rare to fd patients younger than age 20 malignant transfmation.5 Differential diagnosis between cell-rich osteosarcoma benign malignant cell tum is controversial even though histological fdgs such as tum fmation anaplasia osteoclast-like were repted to be imptant f diagnosis osteosarcoma.5,9 Malignant cell tum never produces tum, bone, cartilage manner an osteosarcoma does. Bizarre cell fms are found osteosarcoma, but anaplastic nuclei are usually multilobulated, unlike multucleated osteoclast-like characterize malignant cell tums.5 Clical features tum location such as metaphyseal diaphyseal centerg existence radiographic Codman's triangle, tralesional fluffs, ctical balloong, fat onion-sk-like periosteal reaction also aid osteosarcoma diagnosis.5,9,10 Malignant cell tum usually occurs patients advanced age, i.e., over 60 years, whereas most conventional cell tums occur patients years old.5 This case repts a seventeen-year-old patient. The tum located epimetaphysis to epiphysis. Ctical balloong seen radiographically, although small Codman's triangle tralesional fluffs were apparent. Tum osseous tissue production could be seen metastatic lesions. Radiationduced sarcomatous change can be excluded sht duration after radiation rapy15 occurrence out irradiated areas. Based on se fdgs, fal diagnosis a -cell-rich osteosarcoma. Clical features case provide clues to differentiatg cell-rich osteosarcoma cell tum bone, a relatively common neoplasm occurrg epiphyseal regions patients years.2,16 When we meet patients presentg cell tum terms radiological histological fdgs, especially when y are younger than age at which cell tum usually occurs, we should bear md cell-rich osteosarcoma perfm tensive chemapy surgery. Acknowledgments We are grateful to Drs. Yokoo Hideaki, First Department Pathology, Gunma University Faculty Medice, Rikuo Machami, Department Pathology, Kawakita General Hospital f ir valuable discussions regardg pathological diagnosis. We thank Press Kenneth P.H. Pritzker, Department Pathology Labaty Medice, University Tonto, f his valuable suggestions. We also thank Mr. Kiichi Osawa f his technical assistance makg figures. References 1. Eckardt JJ, Grogan TJ. Giant cell tum bone. Cl Orthop 1986 ; 204 : McGrath PJ. Giant-cell tum bone : An analysis fifty-two cases. J Bone Jot Surg [Br] 1986 ; 54 : Komiya S, Inoue A, Nakashima M, et al. Prognostic facts cell tum bone : A modified histological gradg system useful as a guide to prognosis. Arch Orthop Trauma Surg 1986 ; 105 : Lausten GS, Jensen PK, SchiƒÓdt T, et al. Local cell tumour bone. Longterm follow up 31 cases. Int Orthop 1996 ; 20 : Mirra JM. Giant cell tum. In : Mirra JM, Picci P, Gold RH (eds). Bone Tums. Philadelphia : Lea & Febiger, 1989 : Goldenberg RR, Campbell CJ, Bonfiglio M. Giant-cell tum bone. An analysis two hundred eighteen cases. J Bone Jot Surg [Am] 1970 ; 52 : Cheng JC, Johnston JO. Giant cell tum bone. Prognosis treatment pulmonary. Cl Orthop 1997 ; 338 : Bertoni F, Present D, Sudanese A, et al. Giantcell tum bone pulmonary. Six case repts a review literature. Cl Orthop 1988 ; 237 : Mirra JM. Primary, high-grade tramedullary variants. In : Mirra JM, Picci P, Gold RH (eds) Bone Tums. Philadelphia, Lea & Febiger, 1989 : Sato K, Yamaura S, Iwata H, et al. Giant cellrich osteosarcoma : a case rept. Nagoya J Med Sci 1996 ; 59 : Shozaki T, Hirano T, Watanabe H, et al. Tc (V)-99m dimercaptosuccic acid sctigraphy f

5 151 bone st tissue lesions. Cl Nucl Med 2000 ; 25 : Hasegawa T, Hirose T, Kudo E, et al. Immunophenotypic heterogeneity osteosarcoma. Hum Pathol 1991 ; 22 : Siebenrock KA, Unni KK, Rock MG. Giantcell tum bone metastasizg to lungs. A long-term follow-up. J Bone Jot Surg [Br] 1998 ; 80 : Kay RM, Eckardt JJ, Seeger LL, et al. Pulmonary metastasis benign cell tum bone. Six histologically confirmed cases, cludg one spontaneous regression. Cl Orthop 1994 ; 302 : Rock MG, Sim FH, Unni KK, et al. Secondary malignant -cell tum bone. J Bone Jot Surg [Am] 1986 ; 68 : Campanacci M, Baldi N, Biani S, et al. Giant-cell tum bone. J Bone Jot Surg [Am] 1987 ; 69 :