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The Journal of Bone and Joint Surgery 81:409-13 (1999)
© 1999 The Journal of Bone and Joint Surgery, Inc.

Multifocal, Metachronous Giant-Cell Tumor of the Ulna. A Case Report*

YONG-KOO PARK, M.D.{dagger}, KYUNG NAM RYU, M.D.{dagger}, CHUNG-SOO HAN, M.D.{dagger} and DAE KYUNG BAE, M.D.{dagger}, SEOUL, KOREA

Investigation performed at the Departments of Pathology, Diagnostic Radiology, and Orthopedic Surgery, College of Medicine, Kyung Hee University, Seoul


   Introduction
Top
 Introduction
Case Report
 Discussion
 References
 
Giant-cell tumor of bone apparently was first described by Cooper and Travers5 in 1818. In 1996, the condition was reported to account for approximately 5 percent of all primary bone tumors in a series from the Mayo Clinic34. Multicentric giant-cell tumor of bone has been reported in the literature3,7-9,11,14,15,19,21,24,26-28,32,35. The term multicentric usually refers to the occurrence of a primary bone tumor in two bones or more at approximately the same time—that is, synchronous involvement of two bones or more by the same type of tumor. However, in some of the patients described in previous reports, multifocal involvement of the skeleton occurred over a period of years. We report a rare case of multifocal involvement in which a second giant-cell tumor occurred in the diaphysis of the ulna ten years after the resection of a giant-cell tumor from the distal part of that bone. Because of disagreement about whether the lesions represented a multicentric giant-cell tumor, we characterized this as a case of multifocal, metachronous giant-cell tumor of the ulna.


   Case Report
Top
 Introduction
 Case Report
Discussion
 References
 
In 1985, a twenty-five-year-old man noted a painful, palpable mass in the region of the styloid process of the left ulna. Radiographs revealed an osteolytic lesion with indistinct cortical margins involving the distal epiphysis and metaphysis (Fig. 1). A tentative diagnosis of giant-cell tumor was made, and the distal end of the ulna was resected. The resected segment was five centimeters long and three centimeters wide. Histologically, the lesion was composed of uniformly scattered giant cells and spindle-shaped stromal cells arranged in a storiform pattern (Fig. 2). There was also some infiltration by foam cells.



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  		FIG1: Fig. 1 Anteroposterior radiograph, made in 1985, showing an osteolytic lesion in the distal epiphysis and metaphysis of the left ulna, with cortical destruction.

 


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  		FIG2: Fig. 2 Photomicrograph of a section of the lesion, showing multinucleated giant cells and mononuclear stromal cells (hematoxylin and eosin, x 100).

 
Ten years later, in 1995, the patient noted discomfort and tenderness in the middle of the left forearm; the symptoms lasted for two weeks. Radiographs revealed an osteolytic lesion with cortical thinning in the diaphysis of the ulna; no other lesions were seen proximally or distally (Fig. 3-A). Magnetic resonance imaging revealed that the lesion demonstrated low signal intensity on T1-weighted images (Fig. 3-B) and heterogeneous signal intensity on T2-weighted images (Fig. 3-C). No other lesions were apparent on the magnetic resonance images. The lesion was curetted, and the cavity was filled with bone chips. The curetted material consisted of 6.5 milliliters of brownish-yellow, soft, friable tissue. Histologically, the lesion was composed primarily of giant cells and spindle-shaped stromal cells arranged in a storiform pattern. There were numerous foam cells, and osteoblastic activity was very prominent. Cystic spaces also were seen; the spaces were filled with blood clots and lined by flattened cells, and there were giant cells in the walls of the cysts. A diagnosis of giant-cell tumor and secondary aneurysmal bone cyst was made.



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  		FIG3-A: Figs. 3-A, 3-B, and 3-C: Imaging studies, made in 1995 (ten years after the initial operation), demonstrating an osteolytic lesion in the diaphysis of the left ulna. Fig. 3-A: Anteroposterior radiograph demonstrating an osteolytic lesion in the diaphysis and an absence of recurrence at the site of the previous resection in the distal region of the ulna.

 


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  		FIG3-B: Fig. 3-B: T1-weighted coronal magnetic resonance image (repetition time, 450 milliseconds; echo time, twelve milliseconds) showing a diffuse low signal intensity in the region of the lesion and mild cortical expansion.

 


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  		FIG3-C: Fig. 3-C T2-weighted image (repetition time, 3600 milliseconds; echo time, ninety milliseconds) showing heterogeneous signal intensity in the region of the lesion. There was no evidence of an extraosseous lesion.

 
Two years later, in 1997, the patient again noted discomfort in the left arm. A radiograph showed a small osteolytic lesion in the distal aspect of the shaft of the ulna; the lesion had a distinct margin and measured two by one by one centimeter (Fig. 4). The distal end of the ulna was resected; the resected segment was 4.8 centimeters long and 1.4 centimeters wide. Its cut surface showed cystic spaces filled with yellowish, turbid fluid (Fig. 5). Histologically, the lesion was shown to contain variably sized cystic spaces lined by flattened cells. Fibrous tissue, giant cells, and spindle-shaped stromal cells also were present in the walls of the cysts. Again, a diagnosis of giant-cell tumor and secondary aneurysmal bone cyst was made.



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  		FIG4: Fig. 4 Anteroposterior radiograph, made in 1997 (two years after the second operation), showing a small osteolytic lesion in the distal part of the shaft of the left ulna.

 


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  		FIG5: Fig. 5 Photomicrograph of the cut surface of the resected specimen, showing numerous cystic spaces with evidence of trabeculation.

 


   Discussion
Top
 Introduction
 Case Report
 Discussion
References
 
Multicentric giant-cell tumor is extremely rare; our review of the literature revealed only fifty-eight instances of this lesion1-19,21,22,24-29,31-39. Data regarding age and gender were available for fifty-one individuals, including twenty-four male patients and twenty-seven female patients ranging in age from ten to sixty-two years (mean, twenty-four years). We could detect no differences between unicentric and multicentric tumors with regard to either variable. The most common sites of occurrence of multicentric giant-cell tumor were the small bones of the hands, followed by the tibia, femur, radius, and humerus. Involvement of the sphenoid bone and the skull was unusual, and there were no reports of involvement of the thoracic, lumbar, or sacral vertebrae. Both multicentric and unicentric giant-cell tumors typically involved the ends of the long bones, but the former were more likely to involve the metaphysis28.

A number of previous studies have indicated that multicentric giant-cell tumor may be either synchronous (characterized by lesions that are remote from one another but that are discovered within a short period of time and at similar stages of development) or metachronous (characterized by lesions that occur at different times and in different locations)36. Metachronous tumors may be metastatic or may represent a second, independent focus of disease36. Because of disagreement as to whether the case of our patient should have been classified as an instance of multicentric giant-cell tumor, we characterized it as an instance of multifocal, metachronous giant-cell tumor.

Multicentric involvement of a single bone is extremely rare; our review of the English-language literature revealed only nine patients who had a multicentric giant-cell tumor involving a single bone2,10,13,28,29,31. Five patients had involvement of the tibia, three had involvement of the femur, and one had involvement of the radius. The mean interval between the occurrence of the first and second lesions was 1.5 years. There were two male patients (twenty and forty-three years old) and seven female patients (eleven to fifty-three years old). In 1994, Hindman et al. reported the case of an eleven-year-old girl who had a multicentric giant-cell tumor involving the tibia13. At the time of presentation the patient had a lesion in the distal portion of the tibia, and ten months later she had two more lesions in the middle and proximal parts of the tibia. It is possible that the case of that patient represented an instance of recurrent primary giant-cell tumor. It is well known that giant-cell tumors may recur if treated with curettage only. Most recurrences develop within two years, and almost all occur within five years. In our patient, the primary giant-cell tumor in the distal end of the ulna was resected with a margin of normal bone. The second lesion occurred ten years later in the diaphysis of the ulna. Thus, the second lesion occurred distant from the margin of resection. If the second lesion had been a recurrent lesion, it would have occurred at the site of the margin of resection. However, because of the previously mentioned difference of opinion regarding the term multicentric, we characterized the lesions in our patient as a multifocal, metachronous giant-cell tumor of the ulna.

Before a diagnosis of multicentric giant-cell tumor can be made, it is necessary to rule out the presence of hyperparathyroidism, which can produce features of a polyostotic osteolytic lesion that are virtually identical to those of a giant-cell tumor of bone. The clinical and laboratory findings associated with hyperparathyroidism include hypercalcemia, hypercalciuria, hypophosphatemia, elevated serum levels of alkaline phosphatase, polydipsia, polyuria, generalized weakness, easy fatigability, hypertension, and disturbances of the gastrointestinal system and the central nervous system.

Other primary osseous lesions also may have a multicentric presentation. Fibrosarcoma of bone is usually a solitary lesion, but in certain rare cases multiple independent tumors develop simultaneously throughout the skeleton30. A review of the experience at the Mayo Clinic revealed that two of 255 patients with fibrosarcoma of bone had a multicentric lesion, six of 1786 patients with osteogenic sarcoma of bone had multiple metachronous lesions, and one of eighty-seven patients with chondroblastoma had two separate lesions34. Rarely, osteoblastoma may present as a multicentric lesion20,23.

Ten years elapsed between the appearance of the first and second lesions in our patient. The patient was in the age-group in which tumors most frequently occur, and the radiographic appearance of the lesion was consistent with a diagnosis of giant-cell tumor at the distal end of the ulna.

NOTE: The authors thank Dr. K. K. Unni of the Mayo Clinic, Rochester, Minnesota, for reviewing this case.


   Footnotes
 
*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.

{dagger}Departments of Pathology (Y.-K. P.), Diagnostic Radiology (K. N. R.), and Orthopedic Surgery (C.-S. H. and D. K. B.), College of Medicine, Kyung Hee University, 1 Hoeki-dong, Dongdaemoon-ku, Seoul 130-702, Korea. E-mail address for Dr. Park: damia@chollian.net.


   References
Top
 Introduction
 Case Report
 Discussion
 References
 

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