ORIGINAL RESEARCH
Fresh-frozen Osteochondral Allograft Reconstruction of a Giant Cell Tumor of the Talus Andrew J. Schoenfeld, MD,1 Mark C. Leeson, MD, FACS,2 and Jordan P. Grossman, DPM, FACFAS3 The use of fresh-frozen osteochondral allografts has been reported for the treatment of talar fractures, osteochondral lesions, and tumors of the calcaneus. Currently, we are unaware of any reports in the literature addressing the use of fresh-frozen osteochondral allograft for the treatment of giant cell tumors in the talus. We report our attempt to eradicate an aggressive giant cell tumor of the talus while minimizing morbidity and loss of function via reconstruction with a fresh-frozen osteochondral allograft. This is the first report in the literature to propose such a treatment option for giant cell tumors in the talus. The patient was informed that a report of this case would be submitted for publication. ( The Journal of Foot & Ankle Surgery 46(3):144 –148, 2007) Key words: giant cell tumor, talus, osteochondral allograft
Giant cell tumor is an extremely rare neoplasm in the talus that represents only 14 cases currently reported in the literature (1– 4). These tumors are distinct entities that arise from mesenchymal cells and connective tissue residing within the bone marrow. Although usually benign, the giant cell tumor can be an extremely aggressive lesion and may metastasize in 3% of patients (5). Besides their predilection for aggressive behavior, these neoplasms are particularly challenging because of their tendency to recur, even after thorough curettage and multiple
Address correspondence to: Andrew J. Schoenfeld, MD, Resident, Department of Orthopaedic Surgery, Akron General Medical Center, 224 West Exchange St, Akron, OH 44302. E-mail:
[email protected]. 1 Clinical Instructor, Department of Orthopaedic Surgery, Northeastern Ohio Universities College of Medicine, Resident, Department of Orthopaedic Surgery, Akron General Medical Center, Akron, OH. 2 Professor and Chair, Department of Orthopaedic Surgery, Northeastern Ohio Universities College of Medicine, Attending Faculty and Chairman, Department of Orthopaedic Surgery, Akron General Medical Center, Akron, OH. 3 Associate Professor, Department of Orthopaedic Surgery, Northeastern Ohio Universities College of Medicine, Residency Director, Department of Foot and Ankle Surgery, Saint Vincent’s Charity Hospital, Cleveland, OH, Attending Faculty, Department of Orthopaedic Surgery, Akron General Medical Center, Akron, OH. Copyright © 2007 by the American College of Foot and Ankle Surgeons 1067-2516/07/4603-0002$32.00/0 doi:10.1053/j.jfas.2006.10.004
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surgeries (1, 3). Current treatment options for giant cell tumor include curettage and grafting, en-bloc resection with grafting or arthrodesis, chemical cautery, talectomy, and amputation (1, 4, 6). Although some of the more aggressive procedures are associated with significant joint morbidity and loss of function, simple curettage and grafting of the lesion is insufficient, with recurrence rates reported as high as 50% to 90%, especially when located in the bones of the hands and feet (4, 6). The use of fresh-frozen osteochondral allografts has been reported for the treatment of talar fractures (7), osteochondral lesions (8 –10), and tumors of the calcaneus (11). Currently, we are unaware of any reports in the literature addressing the use of fresh-frozen osteochondral allograft for the treatment of giant cell tumors in the talus. We report our attempt to eradicate an aggressive giant cell tumor of the talus while minimizing morbidity and loss of function via reconstruction with a fresh-frozen osteochondral allograft. This is the first report in the literature to propose such a treatment option for giant cell tumors in the talus. The patient was informed that a report of this case would be submitted for publication. Case Report A 34-year-old woman presented to a fellowship-trained orthopaedic oncologist (M. C. L.) after 2 failed procedures
FIGURE 2 Photograph showing the operative incision and the talar defect after en-bloc resection of the giant cell tumor. A total volume of 48 cm3 was removed from the talus.
FIGURE 1 Preoperative anteroposterior (A) and lateral (B) radiographs of the ankle demonstrating an aggressive giant cell tumor in the medial aspect of the talus. The volume of the lesion measured 11.88 cm3.
for giant cell tumor of the right talus. The tumor had been diagnosed a year earlier and failed 2 prior attempts at curettage and bone grafting. At the time of presentation to the orthopaedic oncologist, the patient had complaints of significant pain and limitation of ankle, as well as subtalar, motion. Because of the aggressive nature of the lesion, it was suggested that a more substantial surgery than simple curettage and bone grafting was necessary. Nonetheless, given the patient’s young age, it was desired to preserve optimal joint function and decrease morbidity. With these factors in mind, a radical resection of the giant cell tumor followed by reconstruction with an osteochondral allograft
was recommended as the optimal treatment modality. This option allowed for a more aggressive local treatment, removal of microscopic disease, and a reconstruction that would potentially maintain relatively normal biomechanics and function. Before the procedure, plain films (Fig 1, A and B) and a computed tomography (CT) scan of the patient’s ankle and foot were obtained. A chest CT was also ordered for the purposes of tumor staging. The patient’s chest CT was negative for any pathological process or metastases. The CT of the foot and ankle revealed an aggressive giant cell tumor in the medial aspect of the talus, with a volume of 11.88 cm3 (2.75 ⫻ 1.8 ⫻ 2.4 cm). There was no evidence of joint disruption or spread to any of the adjacent bones. A side- and height-matched fresh-frozen talar allograft was obtained from our institution’s tissue bank at the time of surgery. The patient’s 2 prior surgeries had involved a longitudinal medial incision and a medial malleolar osteotomy, and it was decided that the same incision and osteotomy site be used. A 12-cm incision was made along the central portion of the medial malleolus. An oblique osteotomy of the medial malleolus was then performed, allowing the osteotomized fragment to be reflected inferiorly on the hinge of the deltoid ligament. This allowed excellent exposure of the talar dome and medial portion of the talus. The medial aspects of the dome and talar body were visibly abnormal, with soft articular cartilage appreciable by palpation. A 15-blade knife was used to demarcate the margins of the lesion, followed by an osteotome, which completed a significant portion of the en-bloc resection. Aggressive curettage with straight and angled curettes was then performed. The resected margins were copiously irrigated with pulse lavage and debrided with a high-speed burr. In total, VOLUME 46, NUMBER 3, MAY/JUNE 2007
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FIGURE 3 Intraoperative photograph showing the contouring of the osteochondral allograft.
a volume of 48 cm3 (4 ⫻ 4 ⫻ 3 cm) was removed from the talus (Fig 2). The medial portion of the osteoarticular allograft was then resected with an oscillating bone saw (Fig 3). The allograft was fashioned to a size approximately 4 ⫻ 4 ⫻ 3 cm and placed into the defect that had been created during excision of the giant cell tumor. An anatomic reduction was obtained between the graft and the host talus and confirmed with intraoperative fluoroscopy. The allograft was secured into position via 2 partially threaded 3-0 cannulated cancellous screws passed from medial to lateral (Fig 4). The osteotomized medial malleolus was anatomically reduced and fixated with 2 fully threaded cortical screws. The patient was placed in a splint after surgery and converted to a cast in the immediate postoperative period. The patient remained non–weight-bearing on the affected extremity for 3 months, after which she was progressively advanced to full weight-bearing with a brace. The patient was evaluated at 1, 2, 7, 9, and 12 months postoperative. Postoperative radiographs revealed intact hardware and progressive incorporation of the osteochondral allograft (Fig 5, A and B). There was no evidence of hardware failure, osteonecrosis, or recurrence of the giant cell tumor. A CT scan of the ankle joint obtained at 9 months postoperative revealed consolidation of the osteochondral allograft (Fig 6). On physical examination, the patient demonstrated full plantarflexion with dorsiflexion limited to only 90° to 100° (Fig 7, A and B). The patient’s subtalar motion was approximately 80% that of her unaffected side. At more than 1 year after surgery, the patient remains pain free and has returned to work. She is currently fully able to participate in all activities of daily living. 146
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FIGURE 4 Final intraoperative photograph demonstrating the anatomic reconstruction of the talar dome with osteochondral allograft. The graft was fixed to the talus with 2 fully threaded cortical screws.
Discussion Giant cell tumor of the talus is a rare entity that has only been reported a handful of times in the orthopaedic literature (1– 4, 6, 12, 13). Nonetheless, it presents a difficult problem because the lesion is locally destructive, recurs even in the face of aggressive surgery, and, on occasion, may even metastasize. Murari et al found giant cell tumors to be the most common benign primary osseous neoplasms in the foot, comprising 19% of all lesions (12). The tumor is frequently found in the calcaneus and metatarsals, rarely affecting the talus (1, 11). When the tumor does present within the talus, it is most commonly localized to the head and neck (1, 4). Giant cell tumors have most often been reported to affect skeletally mature adults, with the mean age of diagnosis in the late 20s to early 30s (4 – 6, 12). Thus, these lesions affect a comparatively young and healthy patient population, making the aggressive nature of the tumor and potential surgical morbidity all the more devastating, especially when localized to an articular region. Unrecognized or under-treated giant cell tumors can result
FIGURE 6 A postoperative CT reconstruction demonstrating full incorporation of the allograft to the retained portion of the talus.
FIGURE 5 Postoperative anteroposterior (A) and lateral (B) radiographs of the ankle showing incorporation of the allograft and maintenance of the articular surface.
in pathologic fractures and irreparable osseous destruction, and can spread to surrounding bones, joints, soft tissues, liver, and lungs (3). As in the case of the patient in this report, the most common initial intervention consists of curettage and bone grafting. The recurrence rate of giant cell tumor after this procedure has been reported to be as high as 50% to 90% depending on location (6, 13). In their review of 10 cases of giant cell tumor of the talus, Malawer and Vance found no cases of recurrence after curettage with or
without bone grafting, leading these authors to speculate that giant cell tumor of the talus represented a less aggressive variant of the condition (4). Nevertheless, Wold and Swee found a recurrence rate of 75% in giant cell tumors of the tarsal bones after grafting (6), and there has been 1 case reported of a talar giant cell tumor metastasizing to the liver and lungs (3). Wold and Swee found that the mean time to recurrence after curettage and grafting was 6.6 months, with the longest time to recurrence occurring at 11 months in their cohort (6). Once the lesion has been found to recur after curettage and grafting, a variety of treatment options are available to the orthopaedic surgeon. These include cryosurgery, enbloc resection with grafting, radical resection, chemotherapy, talectomy and arthrodesis, as well as amputation (1, 3, 4, 12, 13). Although the prevention of tumor recurrence, further osseous destruction, and metastasis are paramount, the significant morbidity associated with many of the above procedures renders them unattractive to both patient and surgeon. Radical resection of the talar lesion with freshfrozen osteochondral allograft allows for complete eradication of the tumor but also attempts to restore joint biomechanics and preserve function. Fresh-frozen talar osteochondral allograft transplants have been used successfully in the treatment of osteochondral lesions (8 –10) and fractures of the talus (7). Furthermore, Muscolo et al demonstrated their success in treating calcaneal tumors with fresh-frozen allografts (11). Gross et al reported their experience with 9 cases where osteochondral defects of the talus were reconstructed with fresh whole bone allografts (8). At an average of 11 years’ follow-up, 6 of the 9 grafts remained in situ with no eviVOLUME 46, NUMBER 3, MAY/JUNE 2007
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consider this treatment option for our patient, who presented with a large, recurrent giant cell tumor of the talus. This case represents the first report in the literature of a giant cell tumor of the talus treated with fresh-frozen osteochondral allograft. At short-term follow-up, the patient enjoys an excellent result and is able to perform all activities of daily living without pain. Furthermore, there has been no further recurrence of the giant cell tumor, and the biomechanical function of the ankle and subtalar joints has been apparently preserved without evidence of increased morbidity. We feel that a fresh-frozen osteochondral allograft is a viable treatment option for giant cell tumor of the talus that has recurred after curettage and bone grafting. This procedure allows for radical resection of the tumor and microscopic disease, while limiting morbidity and attempting to preserve foot biomechanics and function. Although early results are encouraging, longer follow-up and a larger number of subjects are necessary before a full assessment of the capabilities and limitations of this procedure can be made. References
FIGURE 7 Postoperative photographs demonstrating the patient’s ankle range of motion. At 1-year follow-up, the patient demonstrated full plantarflexion (A) with dorsiflexion limited to 90° to 100° (B).
dence of postoperative morbidity or osteoarthritic degeneration. Raikin reported 6 cases of osteochondritis dissecans (average lesion volume of 4.38 cm3) treated with press-fit talar allografts and Acutrak screws (Acumed, Hillsboro, OR) (10). At an average follow-up of 23 months, 5 of the 6 grafts remained in situ, leading this author to conclude that osteochondral allografts serve as a viable treatment option for patients with massive osteochondral lesions (10). The use of osteochondral allografts decreases patient morbidity, reduces surgical time, and allows for the anatomically accurate resurfacing of larger defects. Potential disadvantages include the risk of disease transmission, immune response, and slower incorporation of the graft (9). The success reported in the literature for talar lesions treated with fresh-frozen osteochondral allografts led us to
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1. Kinley S, Wiseman F, Wertheimer SJ. Giant cell tumor of the talus with secondary aneurysmal bone cyst. J Foot Ankle Surg 32:38 – 46, 1993. 2. Lewis MM, Chekofsky K, Wolpin M, Rottderdam HZ. Giant cell tumor of the talus. A case report. Bull Hosp Jt Dis Orthop Inst 42:257–260, 1982. 3. Mella O, Dahl O, Bang G, Engedal H, Gothlin J, Lunde OD. Chemotherapy of a malignant, metastasizing giant-cell tumor of bone. Cancer 50:207–211, 1982. 4. Malawer MM, Vance R. Giant cell tumor and aneurysmal bone cyst of the talus: clinicopathological review and two case reports. Foot Ankle 4:235–244, 1981. 5. Heck RK. Benign (occasionally aggressive) tumors of bone. In: Campbell’s Operative Orthopaedics, ed 10, pp 813– 826, edited by ST Canale, Mosby, St. Louis, 2003. 6. Wold EC, Swee RG. Giant cell tumor of the small bones of the hands and feet. Semin Diagn Pathol 3:173–184, 1984. 7. Rubel IF, Carrer A. Fresh-frozen osteochondral allograft reconstruction of a severely fractured talus: a case report. J Bone Joint Surg Am 87:625– 629, 2005. 8. Gross AE, Agnidis Z, Hutchison CR. Osteochondral defects of the talus treated with fresh osteochondral allograft transplantation. Foot Ankle 22:385–391, 2001. 9. Tasto JP, Ostrander R, Bugbee W, Brage M. The diagnosis and management of osteochondral lesions of the talus: osteochondral allograft update. Arthroscopy 19:138 –141, 2003. 10. Raikin SM. Stage VI: massive osteochondral defects of the talus. Foot Ankle Clin 9:737–744, 2004. 11. Muscolo DL, Ayerza MA, Aponte-Tinao LA. Long-terms results of allograft replacement after total calcanectomy. J Bone Joint Surg Am 82:109 –112, 2000. 12. Murari IM, Callaghan JJ, Berrey BH, Sweet DE. Primary benign and malignant osseous neoplasms of the foot. Foot Ankle 10:68 – 80, 1989. 13. Goldenberg RR, Campbell CJ, Bonfiglio M. Giant cell tumor of bone: an analysis of 218 cases. J Bone Joint Surg Am 52:619 – 664, 1960.