The Journal of Bone and Joint Surgery 81:703-7 (1999)
© 1999 The Journal of Bone and Joint Surgery, Inc.
Recurrent Giant-Cell Tumor Presenting as a Soft-Tissue Mass. A Report of Four Cases*
FRANCIS YOUNG-IN LEE, M.D. ,
MARK MONTGOMERY, M.D.,
ERIC J. HAZAN, M.D.,
SUZANNE B. KEEL, M.D.,
HENRY J. MANKIN, M.D. and
SUSAN KATTAPURAM, M.D., BOSTON, MASSACHUSETTS
Investigation performed at Massachusetts General Hospital and Harvard Medical School, Boston
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Introduction
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Giant-cell tumor of bone is a benign, locally invasive tumor that has been associated with a rate of local recurrence of 27 percent (forty-one of 151) after intralesional excision and 8 percent (ten of 122) after marginal excision1. The high rate of local recurrence and the occasional development of pulmonary metastasis are manifestations of the locally invasive nature of the tumor4-6,8,9,11,12.
A peripheral rim of ossification has been described as an almost pathognomonic sign of a soft-tissue recurrence2,3,11. However, a soft-tissue recurrence can be difficult to detect, especially when the recurrent lesion is asymptomatic and is not associated with the characteristic ossification.
We report on four patients who had an isolated soft-tissue recurrence of a giant-cell tumor of bone. Although a radiodense peripheral rim of ossification is thought to be pathognomonic of a soft-tissue recurrence of giant-cell tumor, this finding was not apparent on the plain radiographs of any of these patients. In each case, a soft-tissue mass was palpable on physical examination and magnetic resonance imaging scans revealed a soft-tissue mass with a heterogeneous signal pattern. The purpose of this report is to emphasize that a soft-tissue recurrence may not be recognized if a thorough physical examination is not performed and magnetic resonance imaging studies are not carried out.
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Case Reports
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CASE 1. A forty-two-year-old woman was seen by us because of a rapidly enlarging soft-tissue mass in the posteromedial aspect of the proximal part of the right calf. Twenty-six months previously, the patient had been managed with curettage and bone-grafting because of a giant-cell tumor of the proximal aspect of the right tibia. Six months after the initial treatment, the patient had a recurrence of the giant-cell tumor and was managed with curettage and packing with polymethylmethacrylate cement. Ten months later (sixteen months after the diagnosis), the patient had another recurrence and was managed with wide resection followed by reconstruction of the proximal aspect of the tibia with an osteoarticular allograft. In addition, the patient had a wedge resection of the lower lobe of the right lung because of pulmonary metastasis. The patient was followed at another institution postoperatively for ten months, at which time she was evaluated at our institution. Physical examination revealed a nontender soft-tissue mass, nine by seven centimeters in size, that was palpable in the posteromedial aspect of the proximal part of the calf. Plain radiographs showed no evidence of a soft-tissue mass or peripheral circumferential ossification (Fig. 1-A). The allograft was intact. Magnetic resonance imaging studies revealed a heterogeneous soft-tissue mass in the muscle planes of the posteromedial aspect of the proximal part of the calf. The lesion was dark on T1-weighted images (Fig. 1-B) and bright on T2-weighted images (Fig. 1-C). The lesion showed substantial gadolinium enhancement in all areas but its central region, which appeared necrotic. The patient was managed with wide resection of the well circumscribed soft-tissue mass, which measured 7.5 by 5.5 by 4.5 centimeters and was located deep within the posterior compartment (Fig. 1-D). Histological analysis demonstrated a recurrent giant-cell tumor with vascular invasion by tumor cells (Fig. 1-E).
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Figs. 1-A through 1-E: Case 1.
Fig. 1-A: Anteroposterior radiograph of the right tibia, made at the time of resection of the extraosseous recurrent lesion (ten months after resection of the tumor and reconstruction of the proximal aspect of the tibia with an allograft). The allograft is well incorporated.
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Fig. 1-B T1-weighted coronal magnetic resonance imaging scan made through the middle portion of the recurrent mass, showing a well circumscribed lesion with low signal intensity (arrow) within the posteromedial aspect of the proximal part of the calf (repetition time, 600 milliseconds; echo time, eleven milliseconds).
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Fig. 1-C T2-weighted axial magnetic resonance imaging scan showing a mass (arrow) with predominantly high signal intensity and a central area of low signal intensity (repetition time, 5000 milliseconds; echo time, eighty-five milliseconds).
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Fig. 1-D Gross pathological specimen showing a heterogeneous soft-tissue mass with hemorrhagic foci. The mass is well circumscribed by fibrous tissue.
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Fig. 1-E Low-power photomicrograph depicting microscopic foci of bone surrounding the soft-tissue recurrence of the giant-cell tumor (x 125).
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CASE 2. A thirty-six-year-old woman was managed with en bloc resection of a giant-cell tumor involving the proximal part of the right fibula three years before she was seen by us. Eight months after the resection, the patient began to have pain that radiated down the lateral aspect of the right leg. The patient was obese, which made palpation difficult. Plain radiographs revealed a soft-tissue mass without peripheral ossification. Magnetic resonance imaging scans showed a lesion within the fibular head and an associated soft-tissue mass. The patient was managed with complete resection of the proximal aspect of the fibula, which constituted a wide resection of the recurrent tumor. The patient did well for two years, at which time she was first seen by us. A nontender soft-tissue mass, six by five centimeters in size, was palpable at the site of the previous operation. The patient was asymptomatic. Plain radiographs demonstrated a soft-tissue density in the operative bed. A needle biopsy was performed under the guidance of computed tomography, and the findings of histological studies were consistent with a recurrent giant-cell tumor. Radiographs of the chest revealed normal findings. The patient was managed with wide resection of the recurrent tumor.
CASE 3. A fifty-five-year-old man was managed with resection of a giant-cell tumor involving the distal part of the ulna fifteen months before he was seen by us. A round, mobile, nontender, subcutaneous mass was palpated at the operative site five months after the resection. Plain radiographs were unremarkable. The patient was managed with resection of the mass, which was found to be a recurrent giant-cell tumor. When the patient was first seen by us approximately ten months later, two masses were palpable in the soft tissues at the operative site. Plain radiographs showed a single soft-tissue mass with no peripheral calcification, and magnetic resonance imaging scans revealed two separate soft-tissue masses. The first mass, which was located superficial to the extensor carpi ulnaris tendon, was a round, 1.2-centimeter-diameter lesion that demonstrated heterogeneous signal intensity on T2-weighted images as well as mild gadolinium enhancement. The other mass, which was two centimeters in diameter, was located adjacent to the ulnar aspect of the distal part of the radius. The patient was managed with en bloc resection of both soft-tissue masses, which proved to be recurrent giant-cell tumors.
CASE 4. A twenty-seven-year-old woman was seen by us because of recurrence of a giant-cell tumor of the distal aspect of the femur. Twenty months previously, the patient had been managed with curettage of the primary lesion and packing with polymethylmethacrylate cement. The intraosseous tumor had recurred during a pregnancy twelve months after the initial operation. When first seen by us, the patient had a nontender soft-tissue mass, two by two centimeters in size, that was palpable deep to the subcutaneous region adjacent to the previous wound. Plain radiographs revealed no ossification around the soft-tissue mass. Magnetic resonance imaging scans revealed a round, heterogeneous mass, 2.5 centimeters in diameter, in the vastus lateralis muscle. The lesion was dark on T1-weighted images (Fig. 2-A) and bright on T2-weighted images (Fig. 2-B) and demonstrated gadolinium enhancement. The radiographic findings were thought to be most consistent with a benign vascular lesion such as a lymphangioma or a hemangioma. A biopsy was performed under the guidance of computed tomography, and the findings of histological analysis were consistent with a recurrent giant-cell tumor (Fig. 2-C). The patient was managed with curettage of the recurrent intraosseous lesion and en bloc resection of the soft-tissue mass.
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Figs. 2-A, 2-B, and 2-C: Case 4.
Fig. 2-A: T1-weighted coronal magnetic resonance imaging scan of the right thigh, demonstrating a well circumscribed lesion (arrow) in the vastus lateralis muscle (repetition time, 700 milliseconds; echo time, twelve milliseconds).
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Fig. 2-B T2-weighted axial magnetic resonance imaging scan showing a homogeneously bright lesion (arrow) in the vastus lateralis muscle (repetition time, 5300 milliseconds; echo time, seventy-six milliseconds).
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Discussion
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The typical radiographic appearance of an intraosseous giant-cell tumor is a well delineated, eccentric, lucent epiphyseal lesion with a nonsclerotic border abutting the articular surface. Although we have observed that approximately 40 to 50 percent of giant-cell tumors show variable amounts of osteoid and woven bone on histopathological examination, these foci may be small and inconspicuous on radiographs7. Whereas intraosseous recurrence after curettage typically is associated with osteolytic changes at the polymethylmethacrylate cement-host bone interface, extraosseous soft-tissue recurrence may or may not be visible on plain radiographs. Cooper et al. reported seventeen instances of soft-tissue recurrence in their review of 1100 cases of giant-cell tumor2. A peripheral rim of ossification was noted around all but one of the recurrent soft-tissue tumors. Nonossified soft-tissue masses were not included in that series. Other studies2,3,9,11 have also demonstrated rim-like ossification surrounding a soft-tissue recurrence of giant-cell tumor, and this phenomenon is thought to be almost pathognomonic of recurrence.
A peripheral rim of ossification was not apparent on the radiographs of any of the patients described in the present report. In the first patient (Case 1), plain radiographs did not show any evidence of a mass or ossification within the soft tissues. Histopathological examination revealed a few foci of ossification within the tumor as well as at the periphery of the lesion, but peripheral ossification was not detectable on radiographs. The patient's history of pulmonary metastasis and the invasion of the vasculature by tumor cells warranted resection of the recurrent giant-cell tumor; however, the resection was not carried out until the soft-tissue mass had become very large. The recurrent tumors in two patients (Cases 3 and 4) were small, and it was clinically difficult to determine whether the masses represented recurrence of tumor within the soft tissues. Neither of these patients had pain or tenderness in association with the recurrent mass. In two patients (Cases 2 and 3), the soft-tissue recurrence occurred within twelve months after resection of the primary mass. Although none of the patients had a distinct rim of ossification on plain radiographs, three patients (Cases 1, 2, and 3) had some evidence of ossification on histological examination. The same three patients (Cases 1, 2, and 3) had soft-tissue recurrence even after partial resection of the involved bone. One patient (Case 4) had an extraosseous recurrence during pregnancy. Simon et al. reported that either symptoms or the stage of the disease was affected in at least six of thirteen patients in whom a giant-cell tumor was diagnosed during pregnancy10.
The follow-up evaluation after operative treatment of giant-cell tumor usually includes clinical examination and plain radiographs of the involved bone. Radiographs of the chest should be made as well. The operative site should be palpated carefully, as it is during the follow-up evaluation of patients who have a soft-tissue tumor. If plain radiographs demonstrate a soft-tissue mass with a peripheral rim of ossification, a diagnosis of soft-tissue recurrence of giant-cell tumor must be strongly considered2,3,11. If a palpable soft-tissue mass is not apparent on plain radiographs, additional diagnostic studies, particularly magnetic resonance imaging scans or a biopsy performed under the guidance of computed tomography, are indicated to exclude the possibility of extraosseous soft-tissue recurrence.
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Footnotes
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*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. No funds were received in support of this study.
Orthopaedic Oncology Unit, Orthopaedic Surgical Service (F. Y.-I. L., E. J. H., and H. J. M.), Department of Radiology (M. M. and S. K.), and Department of Pathology (S. B. K.), Massachusetts General Hospital, Boston, Massachusetts 02114. E-mail address for Dr. Mankin: hmankin@partners.org.
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References
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Introduction
Case Reports