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Giant cell tumor of tendon sheath with intraosseous invasion: A case report
Giant Cell Tumor of Tendon Sheath With lntraosseous Invasion: A Case Report Kevin C. Booth, MD, G. Stewart Campbell, MD, Donald R. Chase, MD, Loma/inda, CA
Giant cell tumor of tendon sheath (GCTTS) is a common fibrohistiocytic tumor that frequently involves periarticular soft tissues and in certain cases may erode adjacent osseous surfaces. Although this clinically benign tumor may be quite locally aggressive, intraosseous extension is a most uncommon finding. We describe a classic GCTTS that, at the time of presentation, had the uncharacteristic x-ray film appearance of a lyric osseous lesion.
Case Report A 45-year-old man presented with a nontender thumb mass that had gradually increased in size for 3 months. Upon examination, a firm mass 1.5 cm in diameter was found in the thumb pulp space and an additional, separate nodule 0.5 cm in diameter was found to overlie the flexor pollicis longus tendon at the level of the midproximal phalanx. X-ray films showed a well-demarcated, predominantly intraosseous, lytic process involving the metaphyseal base of the distal thumb phalanx, as well as an adjacent soft tissue mass (Figs. 1, 2). An incisiGnal biopsy was performed and the classic changes of a GCTTS were found (Figs. 3, 4). Under different
From the Departments of Orthopaedic Surgery and Pathology and Human Anatomy, Loma Linda University, School of Medicine, Loma Linda, CA. Received for publication Aug. 16, 1994; accepted in revised form March 17, 1995. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Reprint requests: G. Stewart Campbell, MD, Department of Orthopedic Surgery, Loma Linda University Medical Center, 11234 Anderson Street, Room A519, Loma Linda, CA 92350.
1000
The Journal of Hand Surgery
circumstances, an en bloc excision would have been done, but we attempted to preserve maximal dexterity. A relatively more conservative excision and curettage of the tumor were performed. The postcurettage bone was lavaged with 0.25% phenol and packed with cancellous bone from the iliac crest. In the 12 months following the surgery, the wound healed well and follow-up x-ray films showed incorporation of the bone graft. There has been no clinical recurrence of the tumor, and the patient has resumed typing.
Discussion Giant cell tumors of tendon sheath are recognized as the most commonly occurring solid soft tissue tumors of the hand,and are second only to ganglion cysts as the most commonly encountered hand mass. I The tumor is classified as being of fibrohistiocytic origin and is morphologically remarkable for its tendency to show a spectrum of changes ranging from tumors that are almost purely fibrous to those that are mostly histiocytic. A GCTTS may have many giant cells or few, depending on its mix of fibrous and histiocytic (xanthomatous) elements? Because of this varied morphology, a myriad of terms have developed that are virtually synonymous for GCTTS. These include fibrous histiocytoma, pigmented vil-
lonodular synovitis, xanthomatous giant cell tumor, fibrous xanthoma, and localized nodular tenosynovitis. The term pigmented villonodular synovitis is deeply ingrained in the literature as a largely descriptive phrase for those less-circumscribed GCTTSs that grow more diffusely and incorporate hemosiderin pigment from tumoral hemorrhage, a phenomenon that accounts for their darker gross appearance.
The Journal of Hand Surgery / Vol. 20A No. 6 November 1995
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Figure 3. Photomicrograph showing typical multinucleated giant cells. The incorporated nuclei show characteristics similar to those of the background population of rounded polygonal cells. (Original magnification X 160)
Figure 1. Anteroposterior x-ray film showing lyric involvement of the distal phalanx by a giant cell tumor of the tendon sheath.
Figure 4. Photomicrograph showing juxtaposition of polygonal cells with foamy histiocytes, one of the characteristics shared by tumors of fibrohistiocytic origin. (Original magnification X 200)
Figure 2. Lateral x-ray film showing lyric intraosseous lesion with an overlying soft tissue mass of the distal thumb phalanx. The extrinsic mass appears to have eroded (thinned) the adjacent cortex (arrow).
Although the etiology of GCTTS is not c l e a r - - a n d some authors even question whether the process is indeed neoplastic--it has been associated with many conditions, including trauma, inflammation, lipid metabolism disturbances, and immune-mediated processes? The association with immune-mediated processes is supported to some degree by immune marker studies that show a c o m m o n monocytemacrophage lineage. 4 Clinically, GCTTS is an indolent tumor that tends to present in the third to fifth decades of life, has a slight female predominance, s and frequently affects
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Booth et al. / Giant Cell Tumor of Tendon Sheath
the soft tissues of the digits. Unlike the closely related fibrous histiocytoma of skin ("dermatofibroma"), most GCTTSs are tendon based and rarely involve the dermis. Although they are usually well circumscribed and localized, some may grow expansively and diffusely, invading adjacent structures or eroding their surfaces. The x-ray film appearance of GCTTS is usually subtle, and in about half of the cases consists only of a solitary soft tissue shadow/ However, approximately 8% to 14% of the tumors show pressure indentation or erosion of adjacent b o n e y Less common findings include periosteal reaction (8.3%) and/or calcification (5.5%)/ A GCTTS rarely has been reported to involve the medullary cavity while avoiding cortical expansion. 7 In a review by Moore et al. of 115 case of GCTTS involving the hand, 10 cases of bony indentation or pressure indentation were reported; however, there were no cases of intraosseous expansion. 6 Cystic intraosseous lesions are more common in GCTTS that affect knee, hip, and toe jointsr Interestingly, x-ray film findings are reportedly normal in about 20% of the cases. 1 Treatment of GCTTS remains controversial largely due to recurrence rates, which have been reported as high as 30%. 3 This relatively high rate appears to be secondary to incomplete excision, which sometimes results from the use of conservative surgery to maintain functionY In our patient, who expressed a need for continued dexterity, we opted to remove the soft tissue nodules and curette the bone lesion, irrigating the cavity with 0.25% phenol to enhance tumor kill.
Although there has been no evidence of tumor regrowth at 1-year follow-up evaluation, we continue to monitor the patient. If there is recurrence, he will be offered an en bloc excision with arthrodesis of the interphalangeal joint. Unfortunately, the efficacy of phenol treatment is not proven at this time.
References 1. Karasick D, Karasick J. Giant cell tumor of tendon sheath: spectrum of radiologic findings. Skeletal Radiol 1992;21: 219-24. 2. Enzinger FM, Weiss SW. Benign tumors and tumor like lesions of the synovial tissue. In: Gay SM, ed. Soft tissue tumors. 2nd ed. St. Louis: CV Mosby, 1988:638-52. 3. Fyfe S, Macfarlane A. Pigmented viUonodular synovitis of the hand. Hand 1980;12:179-88. 4. Ushijima M, Hashimoto H, Tsuneyoshi M, Enjojii M. Giant cell tumor of tendon sheath (nodular tenosynovitis). Cancer 1986;57:875-84. 5. Myers BW, Masi AT, Feigenbaum SL. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine 1980;59:223-38. 6. Moore JR, Weiland AJ, Curtis RM. Localized nodular tenosynovitis: experience with 115 cases. J Hand Surg 1984; 9A:412-7. 7. Phalen GS, McCormack L J, Gazale WJ. Giant-cell tumor of tendon sheath (benign synovioma) in the hand: evaluation of 56 cases. Clin Orthop 1959;15:140-51. 8. Rao AS, Vigorita VJ. Pigmented villonodular synovitis (giant cell tumor of tendon sheath and synovial membrane). J Bone Joint Surg 1984;66A:76-94. 9. Savage RC, Mustafa EB. Giant cell tumor of tendon sheath (localized nodular tenosynovitis), Ann Plast Surg 1984;13:205-10.
Giant cell tumor of tendon sheath (GCTTS) is a common fibrohistiocytic tumor that frequently involves periarticular soft tissues and in certain cases may erode adjacent osseous surfaces. Although this clinically benign tumor may be quite locally aggressive, intraosseous extension is a most uncommon finding. We describe a classic GCTTS that, at the time of presentation, had the uncharacteristic x-ray film appearance of a lyric osseous lesion.
Case Report A 45-year-old man presented with a nontender thumb mass that had gradually increased in size for 3 months. Upon examination, a firm mass 1.5 cm in diameter was found in the thumb pulp space and an additional, separate nodule 0.5 cm in diameter was found to overlie the flexor pollicis longus tendon at the level of the midproximal phalanx. X-ray films showed a well-demarcated, predominantly intraosseous, lytic process involving the metaphyseal base of the distal thumb phalanx, as well as an adjacent soft tissue mass (Figs. 1, 2). An incisiGnal biopsy was performed and the classic changes of a GCTTS were found (Figs. 3, 4). Under different
From the Departments of Orthopaedic Surgery and Pathology and Human Anatomy, Loma Linda University, School of Medicine, Loma Linda, CA. Received for publication Aug. 16, 1994; accepted in revised form March 17, 1995. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Reprint requests: G. Stewart Campbell, MD, Department of Orthopedic Surgery, Loma Linda University Medical Center, 11234 Anderson Street, Room A519, Loma Linda, CA 92350.
1000
The Journal of Hand Surgery
circumstances, an en bloc excision would have been done, but we attempted to preserve maximal dexterity. A relatively more conservative excision and curettage of the tumor were performed. The postcurettage bone was lavaged with 0.25% phenol and packed with cancellous bone from the iliac crest. In the 12 months following the surgery, the wound healed well and follow-up x-ray films showed incorporation of the bone graft. There has been no clinical recurrence of the tumor, and the patient has resumed typing.
Discussion Giant cell tumors of tendon sheath are recognized as the most commonly occurring solid soft tissue tumors of the hand,and are second only to ganglion cysts as the most commonly encountered hand mass. I The tumor is classified as being of fibrohistiocytic origin and is morphologically remarkable for its tendency to show a spectrum of changes ranging from tumors that are almost purely fibrous to those that are mostly histiocytic. A GCTTS may have many giant cells or few, depending on its mix of fibrous and histiocytic (xanthomatous) elements? Because of this varied morphology, a myriad of terms have developed that are virtually synonymous for GCTTS. These include fibrous histiocytoma, pigmented vil-
lonodular synovitis, xanthomatous giant cell tumor, fibrous xanthoma, and localized nodular tenosynovitis. The term pigmented villonodular synovitis is deeply ingrained in the literature as a largely descriptive phrase for those less-circumscribed GCTTSs that grow more diffusely and incorporate hemosiderin pigment from tumoral hemorrhage, a phenomenon that accounts for their darker gross appearance.
The Journal of Hand Surgery / Vol. 20A No. 6 November 1995
1001
Figure 3. Photomicrograph showing typical multinucleated giant cells. The incorporated nuclei show characteristics similar to those of the background population of rounded polygonal cells. (Original magnification X 160)
Figure 1. Anteroposterior x-ray film showing lyric involvement of the distal phalanx by a giant cell tumor of the tendon sheath.
Figure 4. Photomicrograph showing juxtaposition of polygonal cells with foamy histiocytes, one of the characteristics shared by tumors of fibrohistiocytic origin. (Original magnification X 200)
Figure 2. Lateral x-ray film showing lyric intraosseous lesion with an overlying soft tissue mass of the distal thumb phalanx. The extrinsic mass appears to have eroded (thinned) the adjacent cortex (arrow).
Although the etiology of GCTTS is not c l e a r - - a n d some authors even question whether the process is indeed neoplastic--it has been associated with many conditions, including trauma, inflammation, lipid metabolism disturbances, and immune-mediated processes? The association with immune-mediated processes is supported to some degree by immune marker studies that show a c o m m o n monocytemacrophage lineage. 4 Clinically, GCTTS is an indolent tumor that tends to present in the third to fifth decades of life, has a slight female predominance, s and frequently affects
1002
Booth et al. / Giant Cell Tumor of Tendon Sheath
the soft tissues of the digits. Unlike the closely related fibrous histiocytoma of skin ("dermatofibroma"), most GCTTSs are tendon based and rarely involve the dermis. Although they are usually well circumscribed and localized, some may grow expansively and diffusely, invading adjacent structures or eroding their surfaces. The x-ray film appearance of GCTTS is usually subtle, and in about half of the cases consists only of a solitary soft tissue shadow/ However, approximately 8% to 14% of the tumors show pressure indentation or erosion of adjacent b o n e y Less common findings include periosteal reaction (8.3%) and/or calcification (5.5%)/ A GCTTS rarely has been reported to involve the medullary cavity while avoiding cortical expansion. 7 In a review by Moore et al. of 115 case of GCTTS involving the hand, 10 cases of bony indentation or pressure indentation were reported; however, there were no cases of intraosseous expansion. 6 Cystic intraosseous lesions are more common in GCTTS that affect knee, hip, and toe jointsr Interestingly, x-ray film findings are reportedly normal in about 20% of the cases. 1 Treatment of GCTTS remains controversial largely due to recurrence rates, which have been reported as high as 30%. 3 This relatively high rate appears to be secondary to incomplete excision, which sometimes results from the use of conservative surgery to maintain functionY In our patient, who expressed a need for continued dexterity, we opted to remove the soft tissue nodules and curette the bone lesion, irrigating the cavity with 0.25% phenol to enhance tumor kill.
Although there has been no evidence of tumor regrowth at 1-year follow-up evaluation, we continue to monitor the patient. If there is recurrence, he will be offered an en bloc excision with arthrodesis of the interphalangeal joint. Unfortunately, the efficacy of phenol treatment is not proven at this time.
References 1. Karasick D, Karasick J. Giant cell tumor of tendon sheath: spectrum of radiologic findings. Skeletal Radiol 1992;21: 219-24. 2. Enzinger FM, Weiss SW. Benign tumors and tumor like lesions of the synovial tissue. In: Gay SM, ed. Soft tissue tumors. 2nd ed. St. Louis: CV Mosby, 1988:638-52. 3. Fyfe S, Macfarlane A. Pigmented viUonodular synovitis of the hand. Hand 1980;12:179-88. 4. Ushijima M, Hashimoto H, Tsuneyoshi M, Enjojii M. Giant cell tumor of tendon sheath (nodular tenosynovitis). Cancer 1986;57:875-84. 5. Myers BW, Masi AT, Feigenbaum SL. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine 1980;59:223-38. 6. Moore JR, Weiland AJ, Curtis RM. Localized nodular tenosynovitis: experience with 115 cases. J Hand Surg 1984; 9A:412-7. 7. Phalen GS, McCormack L J, Gazale WJ. Giant-cell tumor of tendon sheath (benign synovioma) in the hand: evaluation of 56 cases. Clin Orthop 1959;15:140-51. 8. Rao AS, Vigorita VJ. Pigmented villonodular synovitis (giant cell tumor of tendon sheath and synovial membrane). J Bone Joint Surg 1984;66A:76-94. 9. Savage RC, Mustafa EB. Giant cell tumor of tendon sheath (localized nodular tenosynovitis), Ann Plast Surg 1984;13:205-10.