Primary bone sarcomas are usually seen in a young population. Recently, in the treatment of pediatric bone sarcomas, amputation has been abandoned and limb salvage surgery became more popular in many orthopedic oncological centers. Although the best treatment of a malignant bone sarcoma must include wide resection of the tumor, reconstruction of the large bony defect left after wide resection is still a major problem, especially in pediatric patients. Single-barrel or double-barrel free vascularized fibular autograft (FVFG) transfer to large bone defects has been described before, and is a well known and safe biological reconstruction method for such cases 1–7. In some patients, the length of the remaining bone defect is longer than the donor fibular graft length, which is a challenge for many orthopedic surgeons who perform limb salvage surgery. We report on a patient with 7 years of follow-up who was treated for osteosarcoma of the distal femur by wide resection and biological reconstruction with intercalary double FVFGs, placed in a vertical array on top of one another inside a fresh-frozen femoral allograft.
An 8-year-old girl was referred to our clinic with persistent right leg pain and swelling for 3 months. Radiographic examination indicated a typical periosteal reaction and new bone formation adjacent to the bone at the distal femur. A Tru-cut biopsy of the lesion was performed immediately; the histopathologic diagnosis was osteosarcoma. Whole-body bone scan and computed tomography of the thorax were carried out to search for possible skip lesions and metastases; none were found. Following an MRI investigation, the tumor was staged as IIB according to the Enneking classification. She underwent four cycles of neoadjuvant chemotherapy, followed by a preoperative radiological examination including anteroposterior and lateral plain radiograms and MRI for surgical planning (Fig. 1a–d). The surgical plan was to biologically reconstruct the bone defect, which would remain after wide resection of the tumor, by a fresh-frozen femoral allograft and two FVFGs inserted within the allograft. A custom-made plate was prepared for the fixation of the fibulae. From 3 cm below the tip of the trochanter major to the distal femoral physis, 26 cm of the femur was resected (Fig. 2). After resection, the remaining femoral segments were fixed with the custom-made plate, which was prepared before (Fig. 3). A 26 cm fresh-frozen femoral allograft was used as a structural graft to close the gap between the proximal and the distal parts of the remaining femur to support mechanical stability. A wide bone window was created on the femoral allograft. Through the bone window, two FVFGs harvested bilaterally were placed in the medullary canal of the fresh-frozen femoral allograft in a vertical array on top of each other (Fig. 4). For both fibulae, end-to-end vascular anastomoses to muscular branches of femoral vessels were performed separately (Fig. 5). The femoral allograft was fixed by the plate with two screws. The patient was allowed to walk with partial weight bearing on the third day postoperatively. Plain radiographs were taken before the patient was discharged from the hospital (Fig. 6a and b). The preoperative diagnosis of osteosarcoma was confirmed by a histopathological examination of the resected tumor; the tumor margins were clear. The tumoral cell necrosis rate was 95%, owing to neoadjuvant chemotherapy. Bone union was achieved at the ninth month of follow-up and fibular graft hypertrophy was observed at the end of the first year postoperatively. Full weight bearing was allowed at that time (Fig. 7a and b). All implants were removed 4 years after the operation for MRI control. At the seventh year of follow-up, there was neither local recurrence nor metastasis and bone scan showed that the fibular grafts were viable. At the last control examination (7 years after the operation), knee ROM was 0°–70°. At the last follow-up, full-length standing anteroposterior and lateral radiographs were obtained to measure limb length discrepancy (Fig. 8a and b). The length of the operated limb was 10.7 cm (6 cm from the femur and 4.7 cm from the tibia) shorter than the other limb. Malalignment and malorientation tests were carried out according to Paley et al. 8,9: the mechanical axis deviation was 25 mm lateral, the mechanical lateral distal femoral angle was 80°, the anatomic lateral distal femoral angle was 77°, the medial proximal tibial angle was 92°, the lateral distal tibial angle was 82°, the medial proximal femoral angle was 119°, and the femoral neck-shaft angle was 154°. Malalignment and malorientation tests were normal on the lateral plane. Figure 9a and b show the clinical images of the patient at the last follow-up.
The results of biological reconstruction of large bone defects after malignant bone tumor resection are more favorable than nonbiological reconstruction methods such as prosthesis, because of the lower rates of complications and difficulties 10,11. Free vascularized bone graft methods yield better results than nonvascularized bone grafts. Surgical techniques and the results of FVFG reconstruction have been well described before 1,11–16. Taylor et al.17 reported this technique in 1975. Weiland reported this technique to be an effective treatment method for the reconstruction of large bone defects following bone tumor resection 18.
In the literature, there are several studies on tumor resection and FVFG reconstruction 1–5,12,13,15,16. FVFG is the most favorable treatment modality for the reconstruction of large bone defects after tumor resections. However, this technique has variable complication rates of 37 to 80%. The most common complications reported in the literature are fracture of the graft, nonunion, and infection 2–4,11,12. In some patients, a double-barrel fibular graft, a tumor-free autologous fresh-frozen strut graft, and plate combination have been applied to increase the stiffness and stability of the construct 2,6,7,13. Thanks to this mechanically stable combination of the construct, not only early mobility of the patient but also vascular anastomoses was achieved and the union rate was increased. Union time of the construct is variable in the literature. Delayed union of the construct is related to surgical deterioration of the microvascular circulation and neoadjuvant or adjuvant radiotherapy and chemotherapy 2–4,11,12. Another main problem of this method is the discrepancy between graft length and defect size. The longest fibular graft length is 30 cm in the literature but the author reconstructed the defect with one FVFG 13. In children, the length of one fibular graft is usually enough for the reconstruction and cannot replace the defect. We solved this problem by using the vertically arranged double FVFG technique. Limb length discrepancy or residual shortening may appear after reconstruction of the defect or at the skeletal maturity of a child 10,19. Although one of the aims of our technique was to maintain limb length and we did not shorten the limb during the surgery, we had 10.7 cm of shortness of the limb at the skeletal maturity of the child. Although the reasons for 6 cm of the shortness of the femur were distal femoral physeal damage and the surgery itself, 4.7 cm of shortness of the tibia at the unoperated site was caused possibly by about a 1-year period of non-weight-bearing activities. Some authors have reported that fibular graft lengthening using an external fixator is a safe and effective method for correcting limb length inequality 19. In our case, residual shortening developed at the skeletal maturity of the patient. We planned lengthening and deformity correction of the extremity by the Ilizarov technique, but the patient and her family rejected the treatment.
After wide resection of bone tumors, biological reconstruction of bone defects is a more favorable method, especially in children. A construct of vertically oriented double FVFGs used in conjunction with a femur allograft can be a good treatment method for biological reconstruction when the length of a single FVFG is not enough to span the defect.
Conflicts of interest
There are no conflicts of interest.
1. Jupiter JB, Bour CJ, May JW. The reconstruction of defects in the femoral shaft with vascularized transfers of fibular bone. J Bone Joint Surg A. 1987;69:365–374
2. William C, Eward DVM, Vasileios K, Lawrence SL, Brian E. Free vascularized fibular graft reconstruction of large skeletal defects after tumor resection. Clin Orthop Relat Res. 2010;468:590–598
3. Chen CM, Disa JJ, Lee HY, Mehrara BJ, Hu QY, Nathan S, et al. Reconstruction of extremity long bone defects after sarcoma resection with vascularized fibula flaps: a 10 year review. Plast Reconstr Surg. 2007;119:915–924
4. Gebert C, Hillman A, Schwappach A, Hoffmann CH, Hardes J, Kleinheinz J, et al. Free vascularized fibular grafting for reconstruction after tumor resection in the upper extremity. J Surg Oncol. 2006;94:114–127
5. Ghert M, Colterjohn N, Manfrini M. The use of free vascularized fibular grafts in skeletal reconstruction for bone tumors in children. J Am Acad Orthop Surg. 2007;15:577–587
6. Bi ZG, Han XG, Fu CJ, Cao Y, Yang CL. Reconstruction of large limb bone defects with a double barrel free vascularized fibular graft. Chin Med J. 2008;121:2424–2428
7. Jones NF, Swartz WM, Mears DC, Jupiter JB, Grosmaan AS. The ‘double barrel’ free vascularized fibular bone graft. Plast Reconstr Surg. 1988;81:378–385
8. Paley D, Tetsworth K. Mechanical axis deviation of the lower limbs: preoperative planning of uniapical angular deformities of the tibia or femur. Clin Orthop. 1992;280:48–64
9. Paley D, Herzenberg JE, Tetswoth K, McKie J, Bhave A. Deformity planning for frontal and sagittal plane corrective osteotomies. Orthop Clin North Am. 1994;25:425–465
10. Mercuri M, Capanna R, Manfrini M, Bacci G, Picci P, Ruggieri P, et al. The management of malignant bone tumors in children and adolescents. Clin Orthop Relat Res. 1991;264:156–168
11. Zaretski A, Amir A, Meller I, Leshem D, Kollender Y, Barnea Y, et al. Free fibula long bone reconstruction in orthopedic oncology: a surgical algorithm for reconstructive options. Plast Reconstr Surg. 2004;113:1989–2000
12. Rose PS, Shin AY, Bishop AT, Moran SL, Sim FH. Vascularized free fibula transfer for oncologic reconstruction of humerus. Clin Orthop Relat Res. 2005;382:66–74
13. Hsu RW, Wood MB, Sim FH, Chao EYS. Free vascularized fibular grafting for reconstruction after tumor resection. J Bone Joint Surg Br. 1997;79:36–42
14. De Boer HH, Wood MB, Hermans J. Reconstruction of large skeletal defects by vascularized fibular transfers: factors that influenced the outcome of union in 62 cases. Int Orthop. 1990;14:121–128
15. Brown KL. Limb reconstruction with vascularized fibular grafts after bone tumor resection. Clin Orthop Relat Res. 1991;26:64–73
16. Brigman BE, Hornicek FJ, Gebhardt MC, Mankin HJ. Allografts about the knee in young patients with high grade sarcoma. Clin Orthop Relat Res. 2004;421:232–239
17. Taylor GI, Miller GD, Ham FJ. The free vascularized bone graft. A clinical xtension of microvascular techniques. Plast Reconstr Surg. 1975;55:533–544
18. Weilan AJ. Current concepts review: vascularized free bone transplants. J Bone Joint Surg Am. 1981;63:166–169
19. Courvoisier A, Sailhan F, Mary P, Damsin JP. Case report lengthening of a vascularized free fibular graft. Clin Orthop Relat Res. 2009;467:1377–1384