The Journal of Foot & Ankle Surgery xxx (2015) 1–4
Contents lists available at ScienceDirect
The Journal of Foot & Ankle Surgery journal homepage: www.jfas.org
Case Reports and Series
Reconstruction of the Midfoot Using a Free Vascularized Fibular Graft After En Bloc Excision for Giant Cell Tumor of the Tarsal Bones: A Case Report Hitomi Hara, MD, PhD 1, Toshihiro Akisue 2, Teruya Kawamoto, MD, PhD 1, Yasuo Onishi, MD, PhD 2, Hiroyuki Fujioka, MD, PhD 3, Kotaro Nishida, MD, PhD 4, Ryosuke Kuroda, MD, PhD 5, Masahiro Kurosaka, MD, PhD 6, Toshihiro Akisue, MD, PhD 7 1
Research Associate, Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, Kobe, Japan Orthopedist, Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, Kobe, Japan 3 Professor, Hyogo University of Health Sciences School of Rehabilitation, Kobe, Japan 4 Lecturer, Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, Kobe, Japan 5 Associate Professor, Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, Kobe, Japan 6 Professor, Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, Kobe, Japan 7 Professor, Division of Motor Function, Department of Rehabilitation Science, and Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, Kobe, Japan 2
a r t i c l e i n f o
a b s t r a c t
Level of Clinical Evidence: 4
We report the case of a 32-year-old Japanese female with a giant cell tumor of bone involving multiple midfoot bones. Giant cell tumors of bone account for approximately 5% of all primary bone tumors and most often arise at the ends of long bones. The small bones, such as those of the hands and feet, are rare sites for giant cell tumors. Giant cell tumors of the small bones tend to exhibit more aggressive clinical behavior than those of the long bones. The present patient underwent en bloc tumor excision involving multiple tarsals and metatarsals. We reconstructed the longitudinal arch of the foot with a free vascularized fibular graft. At the 2-year followup visit, bony union had been achieved, with no tumor recurrence. Ó 2015 by the American College of Foot and Ankle Surgeons. All rights reserved.
Keywords: free vascularized fibular graft giant cell tumor of bone midfoot reconstruction
Giant cell tumor of bone (GCTB) is a benign, locally aggressive neoplasm. It has been reported that GCT of the small bones carries a greater risk of local recurrence and metastasis than GCT of the long bones. Curettage, with or without adjuvant therapy, bone grafting, irradiation, and en bloc excision have been used in the treatment of primary GCTBs (1). En bloc excision ensures the lowest rate of recurrence (2), but it is difficult to reconstruct bone defects and recover a weightbearing gait after resection of GCTB in the lower extremities. We report a case of GCTB with extensive involvement of multiple midfoot bones in an adult female. We performed an en bloc excision and reconstructed the lateral longitudinal arch of the midfoot with a free vascularized fibular graft. Medial foot arch stability was achieved by arthrodesis. The patient provided written informed consent for publication of this case report and the accompanying images (a copy of the written consent form is available for review on request). Financial Disclosure: None reported. Conflict of Interest: None reported. Address correspondence to: Hitomi Hara, MD, PhD, Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1 Kusunoki-cho, Chuo-ku, Kobe, Hyogo 650-0017, Japan. E-mail address:
[email protected] (H. Hara).
Case Report A 32-year-old Japanese female was referred to our hospital with a 1-year history of swelling and pain over the dorsum of her left foot. She was a housewife with 1 child, and she had rheumatoid arthritis and a body mass index of 18 kg/m2. Plain radiographs and computed tomography of the foot showed the near disappearance of the lateral cuneiform, with invasion of the surrounding bones (intermediate cuneiform, navicular, and cuboid; Fig. 1). Magnetic resonance imaging revealed an expansile lobulated lesion involving multiple tarsal bones and the surrounding soft tissue. The lesion was isointense on T1-weighted images and heterogeneously hypointense or hyperintense on T2-weighted images. After intravenous injection of gadolinium, the lesion enhanced heterogeneously on the T1-weighted images (Fig. 2). High uptake in the midfoot was seen with technetium-99m-hydroxy-methylene-diphosphonate bone scintigraphy. An open biopsy was performed through the dorsum of the foot. The histopathologic appearance was of uniform mononuclear cells mixed with numerous osteoclast-like giant cells that were large and contain many nuclei. No sarcomatoid features, such as the formation of bone or osteoid, were present. The histologic findings
1067-2516/$ - see front matter Ó 2015 by the American College of Foot and Ankle Surgeons. All rights reserved. http://dx.doi.org/10.1053/j.jfas.2015.04.020
2
H. Hara et al. / The Journal of Foot & Ankle Surgery xxx (2015) 1–4
Fig. 1. Oblique plain radiograph (A) and computed tomography images (B,C) of the left foot showing diffuse infiltration of an osteolytic lesion in the midfoot area.
confirmed a GCTB. From the imaging findings, the Campanacci grade (3) (Table) for our patient was defined as grade 3. We performed en bloc resection of the tumor, which had replaced some of the tarsal bones and had infiltrated the surrounding soft tissue. An ipsilateral vascularized fibular graft was harvested with 8.5 cm of fibula. The lateral foot arch was reconstructed with the fibula, extending from
the calcaneus to the fourth and fifth metatarsal bones. The calcaneus was osteotomized and sculptured to a concave portion to fit the fibular graft. The distal side of fibular graft was placed between the fourth and fifth metatarsal bones, which were decorticated at the contact surface. Bone fixation was achieved with cortical screws. Because the instability of the medial foot arch was slight, stability
Fig. 2. Magnetic resonance images showing a tumor involving multiple tarsal bones and surrounding soft tissue.
H. Hara et al. / The Journal of Foot & Ankle Surgery xxx (2015) 1–4 Table Campanacci radiologic grading system for giant cell tumors of bone Grade
Description
1 2 3
Intramedullary lesion confined to bone Thinned, expanded cortex Cortical breakout
was achieved by arthrodesis using a Kirschner wire and corticocancellous bone grafts from the remaining fibula between the medial cuneiform and navicular bone. We did not perform mid-column reconstruction because the stability sufficient and space limitations were present (Fig. 3). End-to-end microanastomosis of the peroneal vessels to the anterior tibial vessels was performed using a 9-0 Proline suture after perfusion of heparinized serum into the vessels. The fibular graft medullary bleeding was checked after releasing the vascular clamps. Postoperatively, the patient used crutches with a short leg splint or a patellar tendon-bearing brace with no weightbearing for 3 months. The callus had grown in a part in the periphery of the fibular graft at 3 months radiographically, and the patient began to gradually increase weightbearing. At 4 months postoperatively, the bone had fused radiographically, and the patient was allowed full weightbearing with an insole. At the 2-year follow-up visit, she had no evidence of recurrence and had returned to her preoperative level of activity without any further complications, including none at the donor site (Fig. 4). The range of motion of the ankle was restored. The American Orthopaedic Foot and Ankle Society midfoot scale score was 90 of 100, and the Musculoskeletal Tumor Society functional score was 23 of 30. Discussion GCTB accounts for approximately 5% of all primary bone tumors and approximately 20% of benign primary bone tumors. GCTB most often arises at the ends of the long bones and is rarely found in the
3
small bones of the hands and feet. According to the bone tumor registry in Japan, GCTB of the foot accounts for only 3% of all GCTB (4). Some investigators have reported an incidence of <2% in the foot. The tendency of small bone GCTs to develop in younger individuals has been reported, with a mean patient age of 25 years (1). The usual barriers to tumor growth are less well-developed in the foot than in other locations. The cortical bone of the midfoot and hindfoot is thin and is perforated by numerous vascular channels. Most aggressive tumors arising in this area, including GCTB, readily penetrate the cortex and become extracompartmental early in their development. The biologic features specific to this location have not been identified; however, Biscaglia et al (2) have suggested that small bone GCTs appeared to behave more aggressively than long bone GCTs. They reported an overall recurrence rate of 30.7% for small bone GCTs, a rate greater than that for long bone GCTs (approximately 20%) (2). The treatment of choice for most GCTBs is aggressive curettage with adjuvant treatment. However, Campanacci grade 3 tumors, which have already destroyed the cortex, tend to recur more often. Therefore, when the defect is large and the joint surface has been destroyed, en bloc excision is strongly recommended. After wide tumor resection, skeletal and soft tissue reconstruction presents a challenge to reconstructive surgeons. The reconstructive options for midfoot resection include various autografts or allografts. The fibula has distinct advantages over other types of grafts for the reconstruction of large defects in the midfoot, because of its similarity to the metatarsals in shape and diameter. Recently, vascularized bone grafts have been proved to have many advantages over nonvascularized transplants. The grafting of nonvascularized bone to large bony defects significantly increases the time required for bone union, especially if the graft is >6 to 7 cm long (5,6). In contrast, bone defects 26 cm in length can be effectively treated with free vascularized fibular grafts (FVFGs) (6). Reconstruction using FVFGs is superior to allograft reconstruction, for which the bony union rates have ranged from 68% to 90% and delayed union rates from 51% to 57% (7). Previous series analyzing FVFG reconstruction of
Fig. 3. (A) Diagram of foot: bold line denotes the proposed margin of osseous excision, with complete excision of the intermediate and lateral cuneiforms and cuboid and partial excision of the navicular and second, third, fourth, and fifth metatarsal bases. (B) A line drawing showing reconstruction of the osseous defect with a free vascularized fibular graft laterally and corticocancellous fibular bone grafts medially. (C) Photograph showing the foot after en bloc excision.
4
H. Hara et al. / The Journal of Foot & Ankle Surgery xxx (2015) 1–4
Fig. 4. Standing anteroposterior (A) and lateral (B) radiographs of the foot at 2 years postoperatively showing bony union of the reconstructed fibular graft laterally and chip bone graft medially.
oncologic defects have reported bony union rates of 86% to 100% and a mean interval to union of 7 to 12 months (8). Although the interval to union is a difficult parameter to measure rigorously, Minami et al (9) reported a mean time to union of 15.5 weeks for 104 vascularized fibular grafts (97 free and 7 pedicle vascularized fibular grafts), although their report did not include the foot in the recipient sites. In previous separate case reports of foot reconstruction using FVFGs, partial weightbearing was allowed at about 3 months after surgery, and radiographic bone union was present after approximately 4 to 6 months, similar to our findings (4 months). Stress fracture is a common complication of FVFGs, with a reported incidence of 7.7% to 22.2%. In particular, rates of lower extremity graft fracture of 25% to 45% have been reported (8–10). This complication can be prevented by initial restriction of weightbearing, the use of supportive devices, and careful radiographic evaluation of graft hypertrophy. de Boer and Wood (10) have stated that a vascularized graft should be protected during the first year, with loading gradually increased to enhance remodeling and graft hypertrophy. The frequency of donor site complications has been reported to range from 15% to 55%. The major donor site complications have included wound complications, pain, stiffness, ankle instability, motor weakness, nerve injury, and, most commonly, claw toe deformity of the great toe. Daniels et al (11) reported a significant deleterious effect on foot and ankle functioning, and patients should be apprised of these risks. The proposed advantages of FVFGs are the availability of a longer graft, a greater resistance to infection, rapid incorporation and osseous union, and progressive bone hypertrophy in response to
loading. We believe that reconstruction using FVFGs, especially for young and active patients, deserves consideration as a method of salvaging a functional foot and avoiding amputation, considering the characteristic 2 longitudinal arches, the thin soft tissue layers, and the eventual weightbearing function. References 1. Unni KK, Inwards CY. Dahlin’s Bone Tumors: General Aspects and Data on 10,165 Cases, ed 6., Lippincott Williams & Wilkins, Philadelphia, 2010, pp. 225–240. 2. Biscaglia R, Bacchini P, Bertoni F. Giant cell tumor of the hand and foot. Cancer 88:2022–2032, 2000. 3. Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am 69:106–114, 1987. 4. JOA Musculoskeletal Tumor Committee. Bone Tumor Registry in Japan, National Cancer Center, Tokyo, 2010. 5. Tharayil J, Patil RK. Salvage of foot with extensive giant cell tumour with transfer of vascularized fibular bone graft. Indian J Plast Surg 44:150–156, 2011. 6. Malizos KN, Zalavras CG, Soucacos PN, Beris AE, Urbaniak JR. Free vascularized fibular grafts for reconstruction of skeletal defects. J Am Acad Orthop Surg 12: 360–369, 2004. 7. Chen CM, Disa JJ, Lee HY, Mehrara BJ, Hu QY, Nathan S, Boland P, Healey J, Cordeiro PG. Reconstruction of extremity long bone defects after sarcoma resection with vascularized fibula flaps: a 10-year review. Plast Reconstr Surg 119: 915–924, 2007. 8. Eward WC, Kontogeorgakos V, Levin LS, Brigman BE. Free vascularized fibular graft reconstruction of large skeletal defects after tumor resection. Clin Orthop Relat Res 468:590–598, 2010. 9. Minami A, Kasashima T, Iwasaki N, Kato H, Kaneda K. Vascularised fibular grafts: an experience of 102 patients. J Bone Joint Surg Br 82:1022–1025, 2000. 10. de Boer HH, Wood MB. Bone changes in the vascularised fibular graft. J Bone Joint Surg Br 71:374–378, 1989. 11. Daniels TR, Thomas R, Bell TH, Neligan PC. Functional outcome of the foot and ankle after free fibular graft. Foot Ankle Int 26:597–601, 2005.