cc Benign Giant Cell Tumor of Bone Metastasizing to Lung Report of n Case Arthur A. Gresen, M.D., David C. Dahlin, M.D., Lowell F. A. Peterson, M.D., and W. Spencer Payne, M.D. ABSTRACT A case of benign metastasizing giant cell tumor is presented. Previous experience shows that this is a relatively favorable lesion not having the grave implications of sarcomatous disease, as evidenced by the good long-term results in our previously reported patient [14] and a number of others reported in the literature. An aggressive surgical approach to pulmonary metastasis of giant cell tumors is warranted because long-term control, if not permanent eradication, can be accomplished by that means. B y definition, malignant tumors possess the ability to spread to distant sites. The concept of metastasizing benign tumors is less clear but certainly is of great prognostic importance. Metastasis of benign giant cell tumors of bone has been well described, mostly in the orthopedic literature. The problem is sufficiently unusual to warrant review and presentation of a case of benign giant cell tumor metastasizing to the lung. A 53-year-old woman was first seen at the Mayo Clinic six weeks after accidentally striking her right wrist on the edge of a bathtub. Because of persistent pain, she saw her physician, who noted roentgenographic evidence of an osteolytic lesion in the distal right radius. When seen at the hiayo Clinic in January, 1969, she had an extensively destructive eroding lesion with a pathological fracture of the distal right radius (Fig. 1). Laboratory studies and roentgenograms of the chest revealed no abnormalities. On January 28, 1969, the distal right radius was resected at a point 1.5 cm. proximal to the lesion. The tumor had completely eroded through the bone and extended into the pronator quadratus muscle, which was also resected. Arthrodesis of the right wrist was performed using bone from the left ilium. Pathological investigation revealed a 5.5 x 3 x 3 cm. benign giant cell tumor arising in the epiphyseal end of the radius (Fig. 2) with the distal medullary cavity of the excised portion of bone being free from tumor. After satisfactory healing had occurred, previously placed Kirschner wires were removed in July, 1969, and a Vitallium screw was removed in May, 1970. Roentgenograms of the chest at this time showed no abnormalities. From the Mayo Clinic and Mayo Foundation, Rochester, Minn. Accepted for publication May 2, 1973. Address reprint requests to the Section of.prtl~lications, Mayo Clinic, Roclics~er. Minn. 55901. VOL. 16, NO. 5, NOVEMBER, 1973 531
GRESEN ET AL. FIG. 1. Roentgenogram of right wrist showing lytic lesion of right radius. During the following year the patient noted increasing pain and swelling in her right wrist. In June, 1971, local recurrence of the giant cell tumor was diagnosed. A chest roentgenogram showed evidence of a 2 x 1% cm. noncalcified nodule in the left midlung field which, in retrospect, had been present in May, 1970, and had now increased threefold in diameter (Fig. 3). Results of repeat laboratory studies remained within normal limits. On July 1, 1971, a recurrent giant cell tumor nodule 4 X 3 X 3 cm. was removed from in and adjacent to the previous bone graft, and a separate nodular implant 4 mm. in diameter at the base of the first metacarpal bone was excised. The involved area in the bone graft was thoroughly curetted, the surgical wound was copiously irrigated with normal saline solution, and the wound was puddled with 5 ml. of normal saline containing 15 mg. of thio-tepa for 15 minutes before closure. On July 2, 1971, a left thoracotomy was performed and two nodules, 1.7 FIG. 2. Benign giant cell tumor arising in distnl right Indiits. (HhE: ~250.) 532 THE ANNALS OF THORACIC SURGERY
CASE REPORT: Metaslasizing Benign Bone Tumor FIG. 3. Roentgenogram of chest showing 2 x I?: cm. nonralcified nodule in left rnirllztng field. and 1.0 cm. in diameter, respectively, were found within the substance of the lower lobe. Each nodule was excised by wedge resection, and a stapling device was used to close the cut surfaces of the lung. This resection was performed during temporary pulmonary arterial and venous occlusion, and circulation was not restored until the nature of the process was confirmed by examination of frozen sections. The pulmonary lesion proved to be metastasis of benign giant cell tumor of bone identical to that removed from the wrist (Fig. 4). No other giant cell tumor was present. Follow-up examination at thirteen months after thoracotomy revealed no evidence of recurrent disease in either the chest or the wrist. FIG. 4. Metastutic nodule within left lung parenchyma. (H&E; ~250.) VOL. 16, NO. 5, NOVEMBER, 1973 533
GKESEN ET AL. Comment Metastasis from benign giant cell tumors of bone occurs rarely. In our prior experience at the Mayo Clinic, of the 195 patients with such lesions who were treated between 1910 and May, 1969, only 1 patient had metastasis. Metastasis from this type of lesion has been known since 1926 [5], and other instances may have been described before that time. Approximately 17 acceptable examples have been reported (1, 2, 4, 6-13, 15-17]. Other examples are not acceptable because the histological appearance, the description of the lesions, or a rapidly progressive downhill course suggests that sarcomatous changes had occurred prior to the appearance of metastatic lesions. Treatment of benign giant cell tumors is complicated by a local recurrence rate of almost 50% irrespective of the use of radiation therapy [13]. Ten percent of these lesions become malignant, with the majority of the malignant changes being associated with the use of prior radiation treatments [3]. At the present time there are no reliable histological means for identifying the benign giant cell tumors that are most likely to recur or for predicting which tumors will become malignant. Our criteria for differentiating benign from malignant giant cell tumors have been recently reviewed [3]. The critical characteristic of malignant giant cell tumor is the frankly sarcomatous nature of stromal elements in part of an otherwise ordinary giant cell tumor or, more often, at the site of a previously diagnosed giant cell tumor. In the previously reported case from the Mayo Clinic [14], the patient was a 61-year-old man with a benign giant cell tumor of the distal left radius which was initially treated by curettage, bone grafting, and irradiation. Eighteen months later a local recurrence was treated with irradiation followed by curettage. Four and one-half years after that, a right lower lobectomy with resection of a portion of the diaphragm was performed to remove an 8 x 7 x 3.5 cm. tumor that was histologically identical to the tumor of the radius. The patient has been followed for an additional eleven years and has had no recurrence. The present case is another example of benign metastasizing giant cell tumor. The explanation usually advanced to explain this metastasis is that the tumor cells are frequently observed in the vascular spaces on microscopical examination. Thus, fragments have broken off and established themselves elsewhere in the body. The main importance in differentiating the truly sarcomatous lesion from this benign metastasiling variant is that the patient can be reassured as to the frequently favorable outlook of the latter, and surgical aggressiveness is justified in attacking metastases of giant cell tumors when they are accessible. Even the benign metastasizing tumors can be fatal if they are located where they cannot be resected, such as in the spine [13]. Resection of sarcomatous metastatic lesions is unlikely to be of lasting benefit to patients because the lesions are 534 THE ANNALS OF THORACIC SURGERY
CASE REPORT: Metaistasizing Benign Bone Tumor usually widespread and are combined with a rapid downhill clinical ccurse, but the possibility of the benign variant of the disease must be considered since that form can be successfully treated by resection. Pulmonary implants should be resected because they can grow and produce symptcms on account of their increased size [14]. With benign implants in the lung or elsewhere, surgical resection may be effective in eradicating all disease on a permanent basis. Spontaneous regression of pulmonary metastasis [7] or regression after moderate radiation therapy [ 171 has been reported, further indicating the desirability of surgical exploration, if only for verification of the histological diagnosis when resectability might be debatable. References 1. Cameron, J. A. P., and Marsden, A. T. H. Malignant osteoclastoma: Report of a case. J. Bone Joint Surg. [Br.] 34:93, 1952. 2. Copeland, M. M., and Geschickter, C. F. Malignant bone tumors: Primary and metastatic. CA 13:232, 1963. 3. Dahlin, D. C., Cupps, R. E., and Johnson, E. W., Jr. Giant-cell tumor: A study of 195 cases. Cancer 25:1061, 1970. 4. Dyke, S. C. Metastasis of the benign $-iant.-cell tumour of bone (osteoclastoma). J. Pathol. Bacteriol. 34:259, 1931. 5. Finch, E. F., and Gleave, H. H. A case of osteoclastoma (myeloid sarcoma, benign giant cell tumour) with pulmonary metastasis. J. Pathol. Bacteriol. 29:399, 1926. 6. Geschickter, C. F., and Copeland, M. M. Classification of bone tumors. null. Hosp. Joint Dis. 12:498, 1951. 7. Goldenberg, R. R., Campbell C. J., antl Bonfiglio, M. Giant-cell tumor of bone: An analysis of two hundred and eighteen cases. J. Bone Joint Surg. [Am.] 52:619, 1970. 8. Haas, A., and Ritter, S. A. Benign giant-cell tumor of femur with embolic metastasis in prepuce of penis. Am. J. Surg. 89:573, 1955. 9. Jaffee, H. L. Giant-cell tumour (osteoclastoma) of bone: Its pathologic delimitation antl the inherent clinical implications. Ann. R. Coll. Surg. Engl. 13:343, 1953. 10. Kimball, R. M., and DeSanto, D. A. Malignant giant-cell tumor of the ulna. J. Bone Joinl Surg. [Am.] 40:1131, 1958. 11. Lasser, E. C., and Tetewsky, H. Metastasizing giant cell tumor: Report of an unusual case with indolent bone and pulmonary metastases. Am. J. Roentgenol. Radium Ther. Nricl. Med. 78:804, 1957. 12. Murphy, W. R., and Ackerman, L. V. Benign and malignant giant-cell tumors of bone: A clinical-pathological evaluation of thirty-one cases. Cancer 9:317, 1956. 13. Ottolenghi, C. E., Schajowicz, F., and Mondolfo, S. Su di un ciiso tli tumore gig-anto-cellulare con metastasi. Arch. Pzllti Chir. Organi MOT/. 4: 111, 1954. 14. ban, P., Dahlin, D. C., Lipscomb, P. R., and Bernatz, P. E. Benign giant cell tumor of the radius with pulmonary metastasis. Mayo Clin. Proc. 39:344, 1964. 15. Pan, P., and MacKinnon, W. B. Benign giant cell turnour of thoracic vertebra with pulmonary metastasis. Can. Med. Assoc.,I. 87: 1026, 1962. 16. Russell, D. S. Malignant osteoclastoma antl the association of malignant osteoclastoma with Paget s osteitis cleformans. J. none,loint Szirg. [Br.] 31:281, 1949. 17. Stargardter, F. L., and Cooperman, L. R. Giant-cell tumor of sacrum with multiple pulmonary metastases and long-term survival. Br. J. Radiol. n.s. 44:976, 1971. VOI,. 16, NO. 5, NOVEMBER, 1973 535