Giant Cell Tumor of Tendon Sheath in the Wrist: MRI, Thallium-201 and Gallium-67 Scintigraphic Findings

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497 Giant Cell Tumor of Tendon Sheath in the Wrist: MRI, Thallium-201 and Gallium-67 Scintigraphic Findings Shuzo OKUDAIRA, Shinobu TAKAHASHI*, Yoshitsugu TSUDA**, Kiichi SUZUKI***, Yoshihiko KOTOURA**** Department of Orthopaedic Surgery, Kyoto City Hospital * Department of Orthopaedic Surgery, Shiga University of Medical Science ** Department of Radiology, Nagahama City Hospital *** Department of Orthopaedic Surgery, Takeda Hospital **** Department of Orthopaedic Surgery, Nagahama City Hospital Address for correspondence: Dr. Y. Kotoura, Department of Orthopaedic Surgery, Nagahama City Hospital, 313 Oinui-cho, Nagahama, Shiga 526-8580, Japan TEL: +81 749 68 2300 FAX: +81 749 65 1259 Abstract Imaging evaluation of two cases of giant cell tumor of tendon sheath in the wrist is reported with emphasis on the scintigraphic findings. Markedly-increased thallium and negative gallium uptakes were noted in both cases and appeared helpful in the diagnosis of this rare condition. INTRODUCTION The giant cell tumor of tendon sheath is a slow-growing benign tumor arising from a tendon sheath and has been designated as various other names, e.g., xanthomatous giant cell tumor, sclerosing hemangioma, benign synovioma, fibrous xanthoma of synovium and tenosynovial giant cell tumor6,16) This tumor is macroscopically characterized by its dark yellow, nodular or globular proliferation of synovial linings and is indistinguishable histopathologically from pigmented villonodular synovitis, which occurs within the joint cavity5). The most common site affected by the tumor is the finger, but occasional occurrence in other tendon sheaths has been reported4 611,16,22,27). When the tumor occurs around larger joints, differential diagnosis from other soft tissue tumors becomes more difficult. This tumor is benign but often recurs following incomplete removal6,7), hence accurate preoperative diagnosis is required for appropriate treatment. The authors report two cases of giant cell tumor of tendon sheath occurring in the wrist, in which a combination of thallium-201 and gallium-67 scintigraphies and MRI provided useful information in its differential diagnosis. Key words: giant cell tumor of tendon sheath, thallium-scintigraphy, gallium-scintigraphy, MRI (accepted 2006.12.18)

498 come larger CASE Case mass fore REPORT 1: A 19-year-old in her left wrist consulting our woman joint clinic. cal examination, had noticed since two years The and occasionally mass a be- had be- mor Plain in the joint just proximal X-ray On physi- a 30 by 50 mm elastic was palpated the wrist painful. palmar-radial to the wrist films demonstrated hard tu- side of crease. an osteolytic Fig. 1 Case 1, a 19-year-old female patient. T1-weighted image (1a) shows a hypointense tumor on the volar aspect of the wrist partly invading into the distal radius. Gadolinium-enhanced image (1 b) shows enhancement of the tumor. Fat-suppressed sagittal T2-weighted images (1c) shows the mass to be of mixed signal intensity and of mottled appearance. Thallium-201 scintigram (1d) shows a markedly-increased uptake sharply corresponding to the tumor site.

499 lesion in the revealed radius ginal a soft sclerosis. (Magnetom Erlangen, radial tissue epiphysis, aminations Fig. 2 distal epiphysis. tumor and the bony Magnetic were Vision: lesion resonance performed Germany). invading Siemens on CT scan showed into inhomogeneously imaging a Medical T1-weighted the had mar- 1.5-T exunit System, images a low signal enhanced gadolinium injection T2-weighted images mixed ance signal intensity intensity a marked after which was intravenous (Fig. 1a, b). Fat-suppressed showed the tumor to be of and of mottled appear- (Fig. 1c). Thallium-201 showed mass, increase scintigraphic in uptake scan correspond- Case 2, a 25-year-old female patient. T1-weighted axial (2a) and coronal (2b) images show a hypointense to intermediate-intense mass at the volar side of the radiocarpal joint. Coronal T2*-weighted sagittal MR image (1c) shows a hyperintense mass with mottled appearance. Thallium-201 scintigram (2d) shows a markedly-increased uptake sharply corresponding to the tumor site.

500 Table 1 Summary of imaging studies n.p.: not performed ing to the tumor site (Fig. id), while gallium-67 scan was negative. Technetium99m-MDP bone scintigraphy showed an increased uptake corresponding to the erosion of the radius. An open biopsy was performed and confirmed the diagnosis of diffuse-form giant cell tumor of tendon sheath. In a definitive surgery, marginal excision of the tumor was performed and the surface of the bony lesion was further devitalized by LASER. The tumor was partially adherent to the tendons and tendon sheaths invaded the wrist joint cavity. but had not The excised tumor was an ovoid, 20 by 30 by 40 mm brownyellow, lobulated mass. Four years having passed postoperatively, the wrist function is normal and there is no evidence of local recurrence. Case 2: A 25-year-old woman consulted for a mass in the palmar-radial side of her right wrist joint. On physical examination, the tumor was elastic hard, immobile and slightly the wrist motion was not limited. films showed no bony lesion. tender, but Plain X-ray On MRI, both T1-weighted images and T2-weighted images showed a hypointense mass (Fig. 2a, b). T2* - weighted images showed the mass to be of hyperintensity and of mottled appearance (Fig. 2c). Thallium-201 scintigraphic scan showed a localized increased uptake in the tumor site (Fig. 2d), but gallium-67 scan was negative. Intraoperatively, the tumor was an ovoid, 30 by 30 by 40 mm, dark yellow, lobulated mass which partially adhered to the surrounding tendons and muscles, and was excised marginally. Pathological examination revealed xanthoma cells and giant cells scattered throughout hyalinized fibrous stroma with some cytoplasmic hemosiderin inclusion, corresponding to the typical histopathological appearance of a giant cell tumor of tendon sheath. Two years having passed postoperatively, the patient shows no evidence of local recurrence. The imaging studies are summarized in Table 1. DISCUSSION The giant cell tumor of tendon sheath is a benign tumor or a tumor-like condition with undefined etiology. Its nature has been variously explained as hyperplastic, neoplastic, arthritic or post-traumatic5). According to Jaf f e et al., this tumor arises from the synovium of a tendon sheath and shares a common origin with pigmented villonodular synovitis of the articular synovium9). It is postulated that the difference of these two conditions is owing to the nature of the surrounding anatomic compartments, i.e., intraarticular or extraarticular structures. Distinction between these two

501 conditions is difficult histopathologically, and MR images have been reported to be similar for both3,8,10,15,23,25)enzinger and Weiss differentiated the extraarticular subset further into localized and diffuse forms5), although this differentiation was based principally on clinical pictures. According to this differentiation, our cases corresponded to the diffuse form which affects large joints and not digits. It is this form of tumors which often becomes the subject of diagnostic and therapeutic similar imaging chracteristics benign and malignant problems because of its conditions. to those of other The giant cell tumor of tendon sheath have been examined in detail by MRI, whereas thallium scintigraphic scan has rarely been performed for this condition. In the present cases, MRI findings included low signal intensity of the tumor on T1-weighted images plus a mixedintensity, mottled pattern on fat-suppressed T2- weighted images. Gadolinium-enhanced T1- weighted images showed inhomogeneous enhancement of the mass. The present cases also demonstrated that thallium-201 scan was sharply sensitive irrespective of whether bone was involved or not, whereas gallium-67 scan was negative. Combination of MRI and these scintigraphic findings will improve the efficacy and accuracy of preoperative diagnosis of this tumor. The low signal intensity observed on both T1- weighted and T2-weighted images on MRI is consistent with the previous reports4,10,16, 17,23) Hemosiderin inclusion within the cells is considered responsible for the hypointensity on T2- weighted images23). Enhancement after gadolinium administration was positive in our cases as reported in the literature3). A mottled appearance with mixed signal intensity within the tumor on fat-suppressed T2-weighted images is considered representative of the nodular or globular prolif erative pattern of the tumor and hence is characteristic for this tumor1,24,28,29) There seems to be no published reports on thallium scan performed for a giant cell tumor of tendon sheath. Thallium-201 scan has been reported to yield positive results in pigmented villonodular synovitis, Ewing sarcoma, malignant fibrous histiocytoma, and other neoplasms2,20,21,26) In our experience, thallium-201 scan has affinity for hemangioma, desmoid tumor, synovial sarcoma and metastatic carcinomas. The presented cases demonstrated that thallium is markedly accumulated in the giant cell tumor of tendon sheath. Uptake of technetium-99m imercaptosuccinic acid (DMSA) in various tumors has been studied in detail18,19). When technetium-dmsa is taken up by an undiagnosed soft tissue tumor while gallium is not, the tumor is considered most probably one of a limited number of tumors including giant cell tumor of tendon sheath, pigmented villonodular synovitis, desmoid tumor and hemangioma12 `14) Thallium-201 scan appears to have similar behavior to Tc-99m DMSA in its accumulation pattern of soft tissue tumors. A larger stock of scintigraphic knowledge combined with MRI findings would add to the accuracy of imaging diagnosis of a giant cell tumor of tendon sheath. REFERENCES 1) Agarwal, P. K., Gupta, M., Srivastava, A., et al.: Cytomorphology of giant cell tumor of tendon sheath. A report of two cases. Acta Cytol., 41:587-589, 1997. 2) Caluser, C., Healey, J., Macapinlac, H., et al.: T1-201 uptake in recurrent pigmented villonodular synovitis. Correlation with threephase bone imaging. Clin. Nucl. Med., 17:751-753, 1992. 3) De Beuckeleer, L., De Schepper, A., De Belder, F., et al.: Magnetic resonance imaging of localized giant cell tumour of the tendon sheath. Eur. J. Radiol., 7:198-201, 1997. 4) Demouy, E. H., Kaneko, K., Bear, H. M., et al.:

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