Original Article
255
Massive Intercalary Reconstruction of Lower Limb after Wide Excision of Malignant Tumors: An Alternative to Amputation or Rotationplasty Seung Pil Jang, MD1
1 Department of Orthopaedic Surgery, Samsung Medical Center,
Sungkyunkwan University School of Medicine, Seoul, Republic of Korea 2 Department of Plastic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
Goo-Hyun Mun, MD, PhD2
Address for correspondence Sung Wook Seo, MD, PhD, Department of Orthopaedic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, 50 Irwon-dong, Gangnam-gu, Seoul 135-710, Republic of Korea (e-mail: [email protected]).
J Reconstr Microsurg 2014;30:255–262.
Abstract
Keywords
► massive intercalary reconstruction ► intercalary limb resection ► limb salvage surgery ► amputation ► rotationplasty
In this study, we present the feasibility of intercalary limb resection and massive reconstruction for malignant tumors of lower extremity. Ten cases of lower extremity malignancies that had undergone concomitant bone (and/or joint) and soft-tissue reconstruction after wide excision exceeding two-thirds of the cross-sectional area of the affected limb were reviewed. All cases were indicated for amputation because of an expansive tumor, hematoma from a pathologic fracture, or previous unplanned excision, with or without critical structure involvement. Bone was reconstructed with either an allograft or a tumor prosthesis. Soft-tissue reconstruction was performed to achieve critical structure and coverage, which was required in all cases. The resection margin was clear in all cases, and no soft-tissue graft failure was encountered. During a mean follow-up of 26 months (range, 9–42 months), no patient developed local recurrence in the resection–reconstruction site. Of the 10 patients, 8 patients were able to walk independently, and two were ambulatory but needed crutch support outdoors. Massive intercalary resection and reconstruction can be an effective treatment option for locally progressed or complicated lower extremity malignancies. Considering patient preference and the fair functional outcomes observed, it may be a useful alternative to amputation or rotationplasty.
Contemporary mainstay of the treatment for extremity malignancy is limb salvage surgery, which currently is performed in more than 90% of cases.1–3 Limb salvage surgery has been proven equivalent to amputation not only oncologically but also functionally,4 and the proportion of the limb salvage surgery continues to extend along with advances in diagnostic imaging, surgical techniques, and adjuvant therapies.5,6 However, there still are barriers to limb salvage, such as too large a tumor extent, involvement of a vital structure, inap-
propriate biopsy or excision, or pathologic fracture.7 Amputation is still a valid oncologic treatment option in such situations when a tumor is locally progressed or complicated.8 Sometimes, amputation is inevitable to achieve a curative tumor excision with a negative resection margin, which is considered a crucial prognostic factor for extremity malignancy.8,9 Nonetheless, amputation has its obvious disadvantages, as patients must concede the unaffected distal part of the
received May 17, 2013 accepted after revision August 8, 2013 published online March 3, 2014
Copyright © 2014 by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA. Tel: +1(212) 584-4662.
DOI http://dx.doi.org/ 10.1055/s-0033-1357273. ISSN 0743-684X.
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Seung Han Shin, MD1 Keun-Ho Lee, MD1 Sung Wook Seo, MD, PhD1
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involved limb. Indeed, amputees report low levels of treatment satisfaction, a poignant sense of loss, and difficulties in social life, especially with the opposite sex.10,11 Limb shortening may be considered a substitute for the amputation, and rotationplasty is a representative procedure for such an intention.12 However, limb shortening has some disadvantages, as it can be applied only to regions above the lower leg and the patient still has to cope with a prosthesis, not to mention the emotional discomfort. Distraction osteogenesis could be considered to save the distal part.13 However, it is limited to a short segment, which is not realistic in cases requiring amputation, and takes a long time, which may delay adjuvant therapies. Therefore, intercalary resection of the involved limb followed by massive musculoskeletal reconstruction preserving limb length can be a desirable alternative. We have been performing this procedure as an alternative to amputation since 2008. The purpose of this study was to review the cases treated by this procedure and to evaluate its effectiveness.
Patients and Methods We retrospectively reviewed 10 patients who had undergone massive intercalary reconstruction after wide resection of a malignant tumor in the lower extremity, between December 2008 and October 2011. The data were collected with the approval of the Samsung Medical Center Institutional Review Board (file number 2012-06-018). Patient characteristics are summarized in ►Table 1, and the mean age at surgery was 23 years (range, 6–61). Four patients had lung metastasis at initial presentation. The term “massive intercalary reconstruction’ here is defined as concomitant bone (and/or joint) and soft-tissue reconstruction after wide excision exceeding two-thirds of the cross-sectional area of the affected limb. All cases could have been considered candidates for amputation because of the extents of the pathologies, that is, of the tumor per se or of the hematoma arising from a pathologic fracture or previous unplanned excision, with or without the involvement of a critical structure (e.g., the femoral artery or the full thickness of knee extensor muscle) capable of leaving the distal portion useless after wide excision.
The reconstruction procedures used are summarized in ►Table 1. Bone was reconstructed in all cases with either an allograft or a tumor prosthesis. When a structural allograft was used, it was fixated to remaining host bone(s) with plates and screws. The Modular Universal Tumour and Revision System (MUTARS; Implantcast GmbH, Buxtehude, Germany) was used for prosthesis replacement to reconstruct a mobile joint in four cases and a fused knee in one case. In the single composite reconstruction case, the proximal femur was reconstructed with MUTARS, which was fixed to a structural allograft replacing the distal femur. Soft-tissue reconstruction was performed to provide critical structures and coverage, and was required in all 10 cases. In cases with a full-thickness segmental defect of the quadriceps femoris, the muscle was reconstructed by attaching a tendinous graft proximally to the remnant quadriceps muscle and distally to the (reconstructed) proximal tibia. When the main artery was resected, the vessel was reconstructed using the contralateral greater saphenous vein, and a chimeric myocutaneous free flap or a skin graft was used for coverage. The free flap was harvested from latissimus dorsi muscle in seven cases and rectus abdominis muscle in one case. The vessels of the free flap were anastomosed with saved host vessels (or its branches) or stumps of resected host vessels, either in end-to-side manner or end-to-end manner. The flap donor site was closed primarily in all cases. After a mean 20 days (range, 10–31 days) of postoperative hospital stay, the patients were either discharged or transferred for chemotherapy.
Adjuvant Therapy Adjuvant therapies are summarized in ►Table 1. Metastatectomy with curative intent was performed in three cases with lung metastasis at diagnosis. One patient with initial lung metastasis responded well to preoperative chemotherapy, and metastatectomy was not performed in this patient. Including this patient, preoperative chemotherapy was performed in eight patients using a combination of several agents. Postoperative chemotherapy was initiated at 2 to 4 weeks after surgery in nine cases, and was not performed in the case with adamantinoma. Radiotherapy to the primary tumor site was performed postoperatively in one case.
Outcome Evaluation Surgical Procedures “En bloc” wide excision of the tumor was performed in all cases, while saving the distal part of the involved limb. Excision secured an adequate safety margin, and included skin scars for biopsy, scars resulting from previous unplanned excisions, and all hyperintense regions in T2-weighted magnetic resonance (MR) or gadolinium-enhanced MR images, which included the hematoma from a pathologic fracture and the tissues affected by previous unplanned excision. Consequently, extra-articular resection was performed when the tumor involved an adjacent joint, and the main artery and the full thickness of the quadriceps femoris muscle were also resected when necessary. Thus, excision left a massive intercalary defect in the involved limb. Journal of Reconstructive Microsurgery
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The validity of wide excision was evaluated by microscopic resection margin status on the final pathology report. Oncologic outcomes including survival, local recurrence, and distant metastasis were investigated and descriptive statistics were calculated. The survival status was defined as continuously disease free (CDF), when there had been no further recurrence or metastasis after surgery, and as no evidence of disease (NED), when there had been local recurrence or metastasis after surgery but none were detectable at final follow-up. Details of complications and treatments during follow-up were obtained by reviewing medical records. Functional outcomes were evaluated using Musculoskeletal Tumor Society (MSTS) scores, Toronto Extremity Salvage Scores (TESS), and walking states at final follow-up visits.
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256
M/11
M/27
M/16
F/61
M/6
F/53
M/15
M/10
M/15
M/14
1
2
3
4
5
6
7
8
9
10
Osteosarcoma
Osteosarcoma
Osteosarcoma
Osteosarcoma
Myxofibrosarcoma
Osteosarcoma
Adamantinoma
Osteosarcoma
Osteosarcoma
Osteosarcoma
Histology
Femur, distal
Tibia, distal
Tibia, distal
Tibia, distal
Tibia, shaft
Femur, shaft
Tibia, shaft
Femur, distal
Femur, distal
Femur, distal
Location
Pathologic fracture þ
þ
Unplanned surgery
þ
þ Lung
Lung
Lung
Lung
Metastasis at diagnosis
Prosthesis (fusion)
Allograft
Allograft
Allograft
Allograft
Composite
Allograft
Prosthesis
Prosthesis
Prosthesis
Bone reconstruction
Femoropopliteal bypass with reversed contralateral GSV and chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM and STSG
Chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM, TDN to femoral nerve
Chimeric TDA perforator flap with LDM
Knee extensor reconstruction (Achilles tendon allograft) Rectus abdominis MC free flap
Knee extensor reconstruction (mesh and iliotibial band autograft)
Knee extensor reconstruction (mesh and iliotibial band autograft)
Soft-tissue reconstruction
Deep femoral artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Femoral artery (1) and vein (1)
Anterior tibial artery (1) and vein (1)
Deep femoral artery (1) and vein (2)
Recipient vessels used for flap
Pre, post
Journal of Reconstructive Microsurgery
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Pre, post
Pre, post
Pre, post
Post
Pre, post
Pre, post
Pre, post
Pre, post
Chemotherapy
Lung
Lung
Lung
Metastatectomy
Abbreviations: GSV, greater saphenous vein; LDM, latissimus dorsi muscle; MC, myocutaneous; STSG, split-thickness skin graft; TDA, thoracodorsal artery; TDN, thoracodorsal nerve.
Gender/ Age (y)
Patient no.
Table 1 Clinical characteristics of 10 patients
þ
Radiotherapy
Massive Intercalary Reconstruction of Lower Limb Shin et al.
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Results Oncologic Outcomes The resection margin was clear in all cases, suggesting that the wide excision was successful. Oncologic outcomes are summarized in ►Table 2. During follow-up (mean, 26 months; range, 9–42 months), no patient developed local recurrence at the resection–reconstruction site. Of the four patients who initially presented with lung metastases, three underwent lung metastatectomy with surgery for the primary tumor. At final follow-up, one of the four was CDF; one had NED after repeated lung metastatectomy; one did not undergo lung metastatectomy and remained alive with the disease; and one succumbed to recurrent lung metastasis (pulmonary insufficiency). Of the six patients without distant metastasis at diagnosis, one patient developed inguinal lymph node metastasis and had NED after metastatectomy. Finally, 8 of the 10 patients were alive without any evidence of the tumor; 1 remained alive with initially detected lung metastasis; 1 succumbed to recurrent lung metastasis.
Complications and Treatment Complications are described in ►Table 2. During follow-up, no soft-tissue graft failure was observed. However, an allograft fracture was detected in one patient who had undergone total replacement of the whole right femur with an allograft-prosthesis composite. This fracture developed in the distal femur at 10 months after surgery, and was treated by an open reduction and internal fixation with bone grafting. One patient with osteosarcoma of the distal femur experienced prosthesis loosening, and was treated with conversion to a total femur prosthesis. Delayed deep infection developed in two patients; one at 4 months and one at 20 months postoperatively. The former was treated by open debridement, and the other was treated by open debridement with a polyethylene liner change and long-term intravenous antibiotics. Infections were controlled successfully in both and no sign or evidence of infection was evident at final follow-up.
Functional Outcomes Functional outcomes are summarized in ►Table 2. The mean MSTS functional score at final follow-up was 19.5 (65%; range, 37–87%), and mean TESS was 67% (range, 35–96%). Of the 10 patients, 8 were able to walk independently without the crutch, and the other 2 were also ambulatory, but needed a crutch outdoors.
Illustrative Case 1 A 10-year-old male student was diagnosed with osteosarcoma in the right distal tibia. The tumor was surrounding the bone and was close to the skin (►Fig. 1A). There was no evidence of distant metastasis. The case was considered a typical indication for below-knee amputation. However, after sufficient discussion with the patient and his parents, intercalary limb resection and massive reconstruction was planned. After neoadjuvant chemotherapy, a segmental limb resection leaving the posterior tibial neurovascular Journal of Reconstructive Microsurgery
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bundle and the Achilles tendon was performed (►Fig. 1B). A distal tibial structural allograft was placed and fixated with plates and screws, and the tibialis anterior tendon was reconstructed with a tibialis anterior tendon allograft (►Fig. 1C). Finally, the remaining soft-tissue defect was reconstructed with a chimeric thoracodorsal artery perforator flap plus latissimus dorsi muscle flap and a split-thickness skin graft (►Fig. 1D). End-to-side anastomosis was performed between the thoracodorsal artery and the recipient posterior tibial artery. Adjuvant chemotherapy was initiated at 2 weeks after surgery, and at 4 months postoperatively the patient could walk without a crutch.
Illustrative Case 2 A 6-year-old boy presented with huge osteosarcoma in the right femoral diaphysis (►Fig. 2A). The patient also had resectable lung metastasis. After neoadjuvant chemotherapy and lung metastatectomy, en bloc resection of the femoral lesion was performed, including the femoral nerve and a full segment of quadriceps muscle, but saving the femoral artery and vein, the sciatic nerve, and a small proportion of hamstring muscles (►Fig. 2B). The femur was reconstructed with a composite using MUTARS for the proximal femur and a structural allograft for the distal femur (►Fig. 2C). Chimerictype thoracodorsal artery perforator flap with latissimus dorsi muscle was then harvested. To reconstruct the knee extensor, latissimus dorsi muscle component was inset with anastomosis of thoracodorsal nerve to the muscular branch of the femoral nerve. Finally, skin coverage was achieved with thoracodorsal artery perforator flap with anastomosis of the thoracodorsal artery to the femoral artery in an end-to-side manner (►Fig. 2D). Adjuvant chemotherapy was initiated at 4 weeks after surgery. At 9 months after surgery, he could actively extend the knee with an extension lag of 15 degrees and walk with a crutch outdoors.
Discussion Our results show that massive intercalary resection and reconstruction can be an effective treatment option for locally progressed or complicated lower extremity malignancies, and that it offers an alternative to amputation. No local recurrence occurred in our patients, which undoubtedly represents the most critical barrier to limb salvage. In addition, our procedure did not substantially delay adjuvant therapy, which has been reported to affect survival adversely for some cancers.14,15 These findings suggest that the procedure could be viewed as being equivalent to amputation from the oncologic perspective, although a further investigation with statistically sufficient population and long-term followup is mandatory. The role of reconstructive plastic surgery is increasing in contemporary treatment for extremity malignancy, reducing the amputation rate.16 Regarding the reconstruction in our series, no immediate graft failure, which could cause additional morbidity and delay adjuvant therapy, was encountered. In terms of functional outcomes, 8 of the 10 patients were able to walk independently and the other 2 were able to
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walk unaided at home. MSTS score and TESS were slightly lower than those reported previously for limb salvage procedures or lower extremity amputations in total, and were similar to those for above-knee amputation.4,17–19 This could be a procedural limitation, as intercalary excision removes most of the functional structure in the tumor segment, and thus, reconstruction cannot restore all functions of the saved distal part. Nonetheless, the patients were emotionally satisfied with their saved limbs, although this was not quantified in the present study. Amputation still comprises a significant proportion of the treatments adopted for malignant tumors of the extremity. Besides the oncologic reliability of amputation, contemporary prostheses offer excellent function, to the extent that running is possible with a lower limb prosthesis.8 However, amputations do have disadvantages, in not only emotional but also practical aspects. One cannot always put a prosthesis on, for example, while taking a shower or bathing, and there may not be time enough to put the prosthesis on in some emergent situations. Rotationplasty is a functionally excellent alternative to amputation, but has similar disadvantages, and cannot be applied to regions below the lower leg. However, our procedure also has some limitations. Although no patient developed local recurrence, metastasis was problematic in our series. Four of the 10 patients presented with distant metastasis at diagnosis, and of these, 3 developed recurrent lung metastasis after metastatectomy or achieving a good chemotherapeutic response, and the other died of the disease. In addition, one patient developed de novo inguinal lymph node metastasis. Considering the correlation between the larger tumor size and the greater metastatic potential,20 tumors indicated for amputation may be vulnerable to metastasis. Moreover, when a patient presents with established metastasis, a surgery itself may be debatable because of the possible limitation of life expectancy. Nonetheless, as modern literature recommends removal of all known sites of metastasis because of survival benefit,8 it depends on the consensus of physician and patient whether to perform a surgery. In this study, one patient with recurrent lung metastasis and one patient with lymph node metastasis had NED at final followup after metastatectomy. Another limitation of our procedure is the complications: structural allograft fracture, prosthesis loosening, and delayed deep infection were all encountered in our cohort. An allograft fracture may occur in up to 27% of cases,21,22 infection in approximately 15%,23 and aseptic loosening in approximately 13.6%24 after limb salvage surgery, and our results are comparable with these reports. In addition, the necessity of additional procedures for limb length discrepancy may be a drawback for growing patients. However, some of them and their patients do prefer a saved limb even after being given detailed information about these situations. Our procedure also has some practically challenging aspects. First, massive intercalary reconstruction is technically demanding. The overall rate of free-flap failure has been reported to be approximately 2 to 14%,25–27 although no failure occurred in our series. A team approach and an experienced microsurgery specialist are essential for a Journal of Reconstructive Microsurgery
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AWD Lung, recur 9 M/14 10
Abbreviations: AWD, alive with disease; BG, bone grafting; CDF, continuous disease free; DOD, died of disease; LN, lymph node; MSTS, Musculoskeletal Tumor Society; NED, no evidence of disease; ORIF, open reduction and internal fixation; TESS, Toronto Extremity Salvage Score.
Independent
Independent
Crutch (outdoors) 35 50
83 77
96 CDF
18
11
M/10
M/15
8
9
CDF M/15 7
20
NED Inguinal LN, new (metastatectomy) 29 F/53 6
CDF
Infection (controlled)
83
Independent 80 87
Independent 50 53
Independent
Crutch (outdoors) 42
58 43 CDF
CDF
32
26
F/61
M/6
4
5
Lung, recur M/16 3
33
DOD
Allograft fracture (ORIF with BG)
37
Independent 54 60
Independent 81 77 Loosening (conversion to total femur prosthesis) M/27 2
43
CDF
Independent 96 80 Infection (controlled) NED Lung, recur (metastatectomy) M/11 1
42
Distant metastasis Local recurrence Follow-up time (mo) Gender/ Age (y) Patient no.
Table 2 Oncologic and functional outcomes of 10 patients
Final status
Complication and treatment
Final MSTS score (%)
Final TESS (%)
Walking
Massive Intercalary Reconstruction of Lower Limb
Massive Intercalary Reconstruction of Lower Limb
Shin et al.
Fig. 1 Illustrative case of a 10-year-old patient with osteosarcoma of the right distal tibia. (A) Preoperative MR scan. The case was thought to be an indication for below-knee amputation. (B) An “en bloc” intercalary resection was performed, leaving the posterior tibial neurovascular bundle and the Achilles tendon, which were confirmed to be free of tumor involvement. (C) A distal tibia structural allograft was placed and fixated with plates and screws, and the tibialis anterior tendon was reconstructed with a tibialis anterior tendon allograft. (D) Finally, the remaining soft-tissue defect was reconstructed with a chimeric myocutaneous thoracodorsal artery perforator free flap and a split-thickness skin graft. At 4 months after surgery, the patient could walk without a crutch.
successful outcome. Second, when a major nerve is involved, and thus should be resected, results may be inconsistent. The success rate of a motor nerve graft has been reported to be 21 to 87%.28,29 However, when the tumor is located in the femur or tibia and involves the sciatic or tibial nerve, a poorer outcome can be anticipated. Fortunately, we did not encounter this situation in our study population. Nevertheless, our procedure could be applied to this situation also, as a good function has been reported even after resection of the sciatic nerve.30 Recently, reports have been issued not only on intercalary bone or soft-tissue reconstruction but also on combined bone and soft-tissue reconstruction using free flaps after wide excision of extremity malignancies.31–35 Despite the conceptual similarity, our procedure differs from them in the extent of the pathology, which indicated amputation in our cohort. In our study, six patients had nearly circumferential involvement of deep fascia and the other four had diffuse enhancement on MR images caused by a pathologic fracture or unplanned surgery. A wide excision in such cases would leave
no cutaneous tissue or a very thin layer that is prone to necrotize. Classically an amputation is indicated in this situation. In our series, we chose intercalary resection of the limb, including circumferential skin in some cases. This procedure reduces concerns of necrosis by reconstructing the region of concern at initial surgery. Furthermore, the unaffected distal part was saved with a normal limb length and an adequate surgical margin was achieved. The limitations of this study include the small and heterogeneous study population and short follow-up time. In our hospital, massive intercalary resection and reconstruction of the lower limb accounts for 5.6% of surgeries (excluding biopsies) for primary extremity malignancy. Future studies with more patients and longer follow-up are required to ensure the oncologic and functional effectiveness of our procedure, although the indication applies to only a small proportion of cases with extremity malignancy. In conclusion, massive intercalary resection and reconstruction of the lower limb produced reasonable short-term local outcomes from an oncologic perspective. The unaffected
Fig. 2 Illustrative case of a 6-year-old patient with osteosarcoma of the right femoral shaft. (A) Preoperative MR scan. The case was thought to be an indication for hindquarter amputation. (B) An “en bloc” resection of the femoral lesion was performed, including the femoral nerve and a full segment of quadriceps muscle, but saving the femoral artery and vein, the sciatic nerve, and a small proportion of hamstring muscles. (C) The femur was reconstructed with a composite using the MUTARS for the proximal femur and a structural allograft for the distal femur. The knee extensor was reconstructed with a neuromuscular free flap with anastomosis of the thoracodorsal nerve and the muscular branch of the femoral nerve. Finally, coverage with a myocutaneous free flap was performed, and the thoracodorsal artery and femoral artery were anastomosed in an end-to-side manner. (D) Postoperative X-ray. At 9 months after surgery, he could actively extend the knee with an extension lag of 15 degrees and walk with a crutch outdoors. Journal of Reconstructive Microsurgery
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15 Wright J, Doan T, McBride R, Jacobson J, Hershman D. Variability in
16
17
Conflict of Interest None. 18
Funding None. 19
Financial Disclosure None. 20
References
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distal part and the length of involved limb can be preserved by the procedure even in cases with a locally advanced or complicated malignancy. Because of patient preference and fair functional outcomes, we believe the procedure should be viewed as a useful alternative for the treatment of malignant tumors otherwise indicated for amputation.
Shin et al.
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255
Massive Intercalary Reconstruction of Lower Limb after Wide Excision of Malignant Tumors: An Alternative to Amputation or Rotationplasty Seung Pil Jang, MD1
1 Department of Orthopaedic Surgery, Samsung Medical Center,
Sungkyunkwan University School of Medicine, Seoul, Republic of Korea 2 Department of Plastic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
Goo-Hyun Mun, MD, PhD2
Address for correspondence Sung Wook Seo, MD, PhD, Department of Orthopaedic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, 50 Irwon-dong, Gangnam-gu, Seoul 135-710, Republic of Korea (e-mail: [email protected]).
J Reconstr Microsurg 2014;30:255–262.
Abstract
Keywords
► massive intercalary reconstruction ► intercalary limb resection ► limb salvage surgery ► amputation ► rotationplasty
In this study, we present the feasibility of intercalary limb resection and massive reconstruction for malignant tumors of lower extremity. Ten cases of lower extremity malignancies that had undergone concomitant bone (and/or joint) and soft-tissue reconstruction after wide excision exceeding two-thirds of the cross-sectional area of the affected limb were reviewed. All cases were indicated for amputation because of an expansive tumor, hematoma from a pathologic fracture, or previous unplanned excision, with or without critical structure involvement. Bone was reconstructed with either an allograft or a tumor prosthesis. Soft-tissue reconstruction was performed to achieve critical structure and coverage, which was required in all cases. The resection margin was clear in all cases, and no soft-tissue graft failure was encountered. During a mean follow-up of 26 months (range, 9–42 months), no patient developed local recurrence in the resection–reconstruction site. Of the 10 patients, 8 patients were able to walk independently, and two were ambulatory but needed crutch support outdoors. Massive intercalary resection and reconstruction can be an effective treatment option for locally progressed or complicated lower extremity malignancies. Considering patient preference and the fair functional outcomes observed, it may be a useful alternative to amputation or rotationplasty.
Contemporary mainstay of the treatment for extremity malignancy is limb salvage surgery, which currently is performed in more than 90% of cases.1–3 Limb salvage surgery has been proven equivalent to amputation not only oncologically but also functionally,4 and the proportion of the limb salvage surgery continues to extend along with advances in diagnostic imaging, surgical techniques, and adjuvant therapies.5,6 However, there still are barriers to limb salvage, such as too large a tumor extent, involvement of a vital structure, inap-
propriate biopsy or excision, or pathologic fracture.7 Amputation is still a valid oncologic treatment option in such situations when a tumor is locally progressed or complicated.8 Sometimes, amputation is inevitable to achieve a curative tumor excision with a negative resection margin, which is considered a crucial prognostic factor for extremity malignancy.8,9 Nonetheless, amputation has its obvious disadvantages, as patients must concede the unaffected distal part of the
received May 17, 2013 accepted after revision August 8, 2013 published online March 3, 2014
Copyright © 2014 by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA. Tel: +1(212) 584-4662.
DOI http://dx.doi.org/ 10.1055/s-0033-1357273. ISSN 0743-684X.
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Seung Han Shin, MD1 Keun-Ho Lee, MD1 Sung Wook Seo, MD, PhD1
Massive Intercalary Reconstruction of Lower Limb
Shin et al.
involved limb. Indeed, amputees report low levels of treatment satisfaction, a poignant sense of loss, and difficulties in social life, especially with the opposite sex.10,11 Limb shortening may be considered a substitute for the amputation, and rotationplasty is a representative procedure for such an intention.12 However, limb shortening has some disadvantages, as it can be applied only to regions above the lower leg and the patient still has to cope with a prosthesis, not to mention the emotional discomfort. Distraction osteogenesis could be considered to save the distal part.13 However, it is limited to a short segment, which is not realistic in cases requiring amputation, and takes a long time, which may delay adjuvant therapies. Therefore, intercalary resection of the involved limb followed by massive musculoskeletal reconstruction preserving limb length can be a desirable alternative. We have been performing this procedure as an alternative to amputation since 2008. The purpose of this study was to review the cases treated by this procedure and to evaluate its effectiveness.
Patients and Methods We retrospectively reviewed 10 patients who had undergone massive intercalary reconstruction after wide resection of a malignant tumor in the lower extremity, between December 2008 and October 2011. The data were collected with the approval of the Samsung Medical Center Institutional Review Board (file number 2012-06-018). Patient characteristics are summarized in ►Table 1, and the mean age at surgery was 23 years (range, 6–61). Four patients had lung metastasis at initial presentation. The term “massive intercalary reconstruction’ here is defined as concomitant bone (and/or joint) and soft-tissue reconstruction after wide excision exceeding two-thirds of the cross-sectional area of the affected limb. All cases could have been considered candidates for amputation because of the extents of the pathologies, that is, of the tumor per se or of the hematoma arising from a pathologic fracture or previous unplanned excision, with or without the involvement of a critical structure (e.g., the femoral artery or the full thickness of knee extensor muscle) capable of leaving the distal portion useless after wide excision.
The reconstruction procedures used are summarized in ►Table 1. Bone was reconstructed in all cases with either an allograft or a tumor prosthesis. When a structural allograft was used, it was fixated to remaining host bone(s) with plates and screws. The Modular Universal Tumour and Revision System (MUTARS; Implantcast GmbH, Buxtehude, Germany) was used for prosthesis replacement to reconstruct a mobile joint in four cases and a fused knee in one case. In the single composite reconstruction case, the proximal femur was reconstructed with MUTARS, which was fixed to a structural allograft replacing the distal femur. Soft-tissue reconstruction was performed to provide critical structures and coverage, and was required in all 10 cases. In cases with a full-thickness segmental defect of the quadriceps femoris, the muscle was reconstructed by attaching a tendinous graft proximally to the remnant quadriceps muscle and distally to the (reconstructed) proximal tibia. When the main artery was resected, the vessel was reconstructed using the contralateral greater saphenous vein, and a chimeric myocutaneous free flap or a skin graft was used for coverage. The free flap was harvested from latissimus dorsi muscle in seven cases and rectus abdominis muscle in one case. The vessels of the free flap were anastomosed with saved host vessels (or its branches) or stumps of resected host vessels, either in end-to-side manner or end-to-end manner. The flap donor site was closed primarily in all cases. After a mean 20 days (range, 10–31 days) of postoperative hospital stay, the patients were either discharged or transferred for chemotherapy.
Adjuvant Therapy Adjuvant therapies are summarized in ►Table 1. Metastatectomy with curative intent was performed in three cases with lung metastasis at diagnosis. One patient with initial lung metastasis responded well to preoperative chemotherapy, and metastatectomy was not performed in this patient. Including this patient, preoperative chemotherapy was performed in eight patients using a combination of several agents. Postoperative chemotherapy was initiated at 2 to 4 weeks after surgery in nine cases, and was not performed in the case with adamantinoma. Radiotherapy to the primary tumor site was performed postoperatively in one case.
Outcome Evaluation Surgical Procedures “En bloc” wide excision of the tumor was performed in all cases, while saving the distal part of the involved limb. Excision secured an adequate safety margin, and included skin scars for biopsy, scars resulting from previous unplanned excisions, and all hyperintense regions in T2-weighted magnetic resonance (MR) or gadolinium-enhanced MR images, which included the hematoma from a pathologic fracture and the tissues affected by previous unplanned excision. Consequently, extra-articular resection was performed when the tumor involved an adjacent joint, and the main artery and the full thickness of the quadriceps femoris muscle were also resected when necessary. Thus, excision left a massive intercalary defect in the involved limb. Journal of Reconstructive Microsurgery
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The validity of wide excision was evaluated by microscopic resection margin status on the final pathology report. Oncologic outcomes including survival, local recurrence, and distant metastasis were investigated and descriptive statistics were calculated. The survival status was defined as continuously disease free (CDF), when there had been no further recurrence or metastasis after surgery, and as no evidence of disease (NED), when there had been local recurrence or metastasis after surgery but none were detectable at final follow-up. Details of complications and treatments during follow-up were obtained by reviewing medical records. Functional outcomes were evaluated using Musculoskeletal Tumor Society (MSTS) scores, Toronto Extremity Salvage Scores (TESS), and walking states at final follow-up visits.
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256
M/11
M/27
M/16
F/61
M/6
F/53
M/15
M/10
M/15
M/14
1
2
3
4
5
6
7
8
9
10
Osteosarcoma
Osteosarcoma
Osteosarcoma
Osteosarcoma
Myxofibrosarcoma
Osteosarcoma
Adamantinoma
Osteosarcoma
Osteosarcoma
Osteosarcoma
Histology
Femur, distal
Tibia, distal
Tibia, distal
Tibia, distal
Tibia, shaft
Femur, shaft
Tibia, shaft
Femur, distal
Femur, distal
Femur, distal
Location
Pathologic fracture þ
þ
Unplanned surgery
þ
þ Lung
Lung
Lung
Lung
Metastasis at diagnosis
Prosthesis (fusion)
Allograft
Allograft
Allograft
Allograft
Composite
Allograft
Prosthesis
Prosthesis
Prosthesis
Bone reconstruction
Femoropopliteal bypass with reversed contralateral GSV and chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM and STSG
Chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM
Chimeric TDA perforator flap with LDM, TDN to femoral nerve
Chimeric TDA perforator flap with LDM
Knee extensor reconstruction (Achilles tendon allograft) Rectus abdominis MC free flap
Knee extensor reconstruction (mesh and iliotibial band autograft)
Knee extensor reconstruction (mesh and iliotibial band autograft)
Soft-tissue reconstruction
Deep femoral artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Posterior tibial artery (1) and vein (1)
Femoral artery (1) and vein (1)
Anterior tibial artery (1) and vein (1)
Deep femoral artery (1) and vein (2)
Recipient vessels used for flap
Pre, post
Journal of Reconstructive Microsurgery
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Pre, post
Pre, post
Pre, post
Post
Pre, post
Pre, post
Pre, post
Pre, post
Chemotherapy
Lung
Lung
Lung
Metastatectomy
Abbreviations: GSV, greater saphenous vein; LDM, latissimus dorsi muscle; MC, myocutaneous; STSG, split-thickness skin graft; TDA, thoracodorsal artery; TDN, thoracodorsal nerve.
Gender/ Age (y)
Patient no.
Table 1 Clinical characteristics of 10 patients
þ
Radiotherapy
Massive Intercalary Reconstruction of Lower Limb Shin et al.
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Results Oncologic Outcomes The resection margin was clear in all cases, suggesting that the wide excision was successful. Oncologic outcomes are summarized in ►Table 2. During follow-up (mean, 26 months; range, 9–42 months), no patient developed local recurrence at the resection–reconstruction site. Of the four patients who initially presented with lung metastases, three underwent lung metastatectomy with surgery for the primary tumor. At final follow-up, one of the four was CDF; one had NED after repeated lung metastatectomy; one did not undergo lung metastatectomy and remained alive with the disease; and one succumbed to recurrent lung metastasis (pulmonary insufficiency). Of the six patients without distant metastasis at diagnosis, one patient developed inguinal lymph node metastasis and had NED after metastatectomy. Finally, 8 of the 10 patients were alive without any evidence of the tumor; 1 remained alive with initially detected lung metastasis; 1 succumbed to recurrent lung metastasis.
Complications and Treatment Complications are described in ►Table 2. During follow-up, no soft-tissue graft failure was observed. However, an allograft fracture was detected in one patient who had undergone total replacement of the whole right femur with an allograft-prosthesis composite. This fracture developed in the distal femur at 10 months after surgery, and was treated by an open reduction and internal fixation with bone grafting. One patient with osteosarcoma of the distal femur experienced prosthesis loosening, and was treated with conversion to a total femur prosthesis. Delayed deep infection developed in two patients; one at 4 months and one at 20 months postoperatively. The former was treated by open debridement, and the other was treated by open debridement with a polyethylene liner change and long-term intravenous antibiotics. Infections were controlled successfully in both and no sign or evidence of infection was evident at final follow-up.
Functional Outcomes Functional outcomes are summarized in ►Table 2. The mean MSTS functional score at final follow-up was 19.5 (65%; range, 37–87%), and mean TESS was 67% (range, 35–96%). Of the 10 patients, 8 were able to walk independently without the crutch, and the other 2 were also ambulatory, but needed a crutch outdoors.
Illustrative Case 1 A 10-year-old male student was diagnosed with osteosarcoma in the right distal tibia. The tumor was surrounding the bone and was close to the skin (►Fig. 1A). There was no evidence of distant metastasis. The case was considered a typical indication for below-knee amputation. However, after sufficient discussion with the patient and his parents, intercalary limb resection and massive reconstruction was planned. After neoadjuvant chemotherapy, a segmental limb resection leaving the posterior tibial neurovascular Journal of Reconstructive Microsurgery
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bundle and the Achilles tendon was performed (►Fig. 1B). A distal tibial structural allograft was placed and fixated with plates and screws, and the tibialis anterior tendon was reconstructed with a tibialis anterior tendon allograft (►Fig. 1C). Finally, the remaining soft-tissue defect was reconstructed with a chimeric thoracodorsal artery perforator flap plus latissimus dorsi muscle flap and a split-thickness skin graft (►Fig. 1D). End-to-side anastomosis was performed between the thoracodorsal artery and the recipient posterior tibial artery. Adjuvant chemotherapy was initiated at 2 weeks after surgery, and at 4 months postoperatively the patient could walk without a crutch.
Illustrative Case 2 A 6-year-old boy presented with huge osteosarcoma in the right femoral diaphysis (►Fig. 2A). The patient also had resectable lung metastasis. After neoadjuvant chemotherapy and lung metastatectomy, en bloc resection of the femoral lesion was performed, including the femoral nerve and a full segment of quadriceps muscle, but saving the femoral artery and vein, the sciatic nerve, and a small proportion of hamstring muscles (►Fig. 2B). The femur was reconstructed with a composite using MUTARS for the proximal femur and a structural allograft for the distal femur (►Fig. 2C). Chimerictype thoracodorsal artery perforator flap with latissimus dorsi muscle was then harvested. To reconstruct the knee extensor, latissimus dorsi muscle component was inset with anastomosis of thoracodorsal nerve to the muscular branch of the femoral nerve. Finally, skin coverage was achieved with thoracodorsal artery perforator flap with anastomosis of the thoracodorsal artery to the femoral artery in an end-to-side manner (►Fig. 2D). Adjuvant chemotherapy was initiated at 4 weeks after surgery. At 9 months after surgery, he could actively extend the knee with an extension lag of 15 degrees and walk with a crutch outdoors.
Discussion Our results show that massive intercalary resection and reconstruction can be an effective treatment option for locally progressed or complicated lower extremity malignancies, and that it offers an alternative to amputation. No local recurrence occurred in our patients, which undoubtedly represents the most critical barrier to limb salvage. In addition, our procedure did not substantially delay adjuvant therapy, which has been reported to affect survival adversely for some cancers.14,15 These findings suggest that the procedure could be viewed as being equivalent to amputation from the oncologic perspective, although a further investigation with statistically sufficient population and long-term followup is mandatory. The role of reconstructive plastic surgery is increasing in contemporary treatment for extremity malignancy, reducing the amputation rate.16 Regarding the reconstruction in our series, no immediate graft failure, which could cause additional morbidity and delay adjuvant therapy, was encountered. In terms of functional outcomes, 8 of the 10 patients were able to walk independently and the other 2 were able to
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walk unaided at home. MSTS score and TESS were slightly lower than those reported previously for limb salvage procedures or lower extremity amputations in total, and were similar to those for above-knee amputation.4,17–19 This could be a procedural limitation, as intercalary excision removes most of the functional structure in the tumor segment, and thus, reconstruction cannot restore all functions of the saved distal part. Nonetheless, the patients were emotionally satisfied with their saved limbs, although this was not quantified in the present study. Amputation still comprises a significant proportion of the treatments adopted for malignant tumors of the extremity. Besides the oncologic reliability of amputation, contemporary prostheses offer excellent function, to the extent that running is possible with a lower limb prosthesis.8 However, amputations do have disadvantages, in not only emotional but also practical aspects. One cannot always put a prosthesis on, for example, while taking a shower or bathing, and there may not be time enough to put the prosthesis on in some emergent situations. Rotationplasty is a functionally excellent alternative to amputation, but has similar disadvantages, and cannot be applied to regions below the lower leg. However, our procedure also has some limitations. Although no patient developed local recurrence, metastasis was problematic in our series. Four of the 10 patients presented with distant metastasis at diagnosis, and of these, 3 developed recurrent lung metastasis after metastatectomy or achieving a good chemotherapeutic response, and the other died of the disease. In addition, one patient developed de novo inguinal lymph node metastasis. Considering the correlation between the larger tumor size and the greater metastatic potential,20 tumors indicated for amputation may be vulnerable to metastasis. Moreover, when a patient presents with established metastasis, a surgery itself may be debatable because of the possible limitation of life expectancy. Nonetheless, as modern literature recommends removal of all known sites of metastasis because of survival benefit,8 it depends on the consensus of physician and patient whether to perform a surgery. In this study, one patient with recurrent lung metastasis and one patient with lymph node metastasis had NED at final followup after metastatectomy. Another limitation of our procedure is the complications: structural allograft fracture, prosthesis loosening, and delayed deep infection were all encountered in our cohort. An allograft fracture may occur in up to 27% of cases,21,22 infection in approximately 15%,23 and aseptic loosening in approximately 13.6%24 after limb salvage surgery, and our results are comparable with these reports. In addition, the necessity of additional procedures for limb length discrepancy may be a drawback for growing patients. However, some of them and their patients do prefer a saved limb even after being given detailed information about these situations. Our procedure also has some practically challenging aspects. First, massive intercalary reconstruction is technically demanding. The overall rate of free-flap failure has been reported to be approximately 2 to 14%,25–27 although no failure occurred in our series. A team approach and an experienced microsurgery specialist are essential for a Journal of Reconstructive Microsurgery
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AWD Lung, recur 9 M/14 10
Abbreviations: AWD, alive with disease; BG, bone grafting; CDF, continuous disease free; DOD, died of disease; LN, lymph node; MSTS, Musculoskeletal Tumor Society; NED, no evidence of disease; ORIF, open reduction and internal fixation; TESS, Toronto Extremity Salvage Score.
Independent
Independent
Crutch (outdoors) 35 50
83 77
96 CDF
18
11
M/10
M/15
8
9
CDF M/15 7
20
NED Inguinal LN, new (metastatectomy) 29 F/53 6
CDF
Infection (controlled)
83
Independent 80 87
Independent 50 53
Independent
Crutch (outdoors) 42
58 43 CDF
CDF
32
26
F/61
M/6
4
5
Lung, recur M/16 3
33
DOD
Allograft fracture (ORIF with BG)
37
Independent 54 60
Independent 81 77 Loosening (conversion to total femur prosthesis) M/27 2
43
CDF
Independent 96 80 Infection (controlled) NED Lung, recur (metastatectomy) M/11 1
42
Distant metastasis Local recurrence Follow-up time (mo) Gender/ Age (y) Patient no.
Table 2 Oncologic and functional outcomes of 10 patients
Final status
Complication and treatment
Final MSTS score (%)
Final TESS (%)
Walking
Massive Intercalary Reconstruction of Lower Limb
Massive Intercalary Reconstruction of Lower Limb
Shin et al.
Fig. 1 Illustrative case of a 10-year-old patient with osteosarcoma of the right distal tibia. (A) Preoperative MR scan. The case was thought to be an indication for below-knee amputation. (B) An “en bloc” intercalary resection was performed, leaving the posterior tibial neurovascular bundle and the Achilles tendon, which were confirmed to be free of tumor involvement. (C) A distal tibia structural allograft was placed and fixated with plates and screws, and the tibialis anterior tendon was reconstructed with a tibialis anterior tendon allograft. (D) Finally, the remaining soft-tissue defect was reconstructed with a chimeric myocutaneous thoracodorsal artery perforator free flap and a split-thickness skin graft. At 4 months after surgery, the patient could walk without a crutch.
successful outcome. Second, when a major nerve is involved, and thus should be resected, results may be inconsistent. The success rate of a motor nerve graft has been reported to be 21 to 87%.28,29 However, when the tumor is located in the femur or tibia and involves the sciatic or tibial nerve, a poorer outcome can be anticipated. Fortunately, we did not encounter this situation in our study population. Nevertheless, our procedure could be applied to this situation also, as a good function has been reported even after resection of the sciatic nerve.30 Recently, reports have been issued not only on intercalary bone or soft-tissue reconstruction but also on combined bone and soft-tissue reconstruction using free flaps after wide excision of extremity malignancies.31–35 Despite the conceptual similarity, our procedure differs from them in the extent of the pathology, which indicated amputation in our cohort. In our study, six patients had nearly circumferential involvement of deep fascia and the other four had diffuse enhancement on MR images caused by a pathologic fracture or unplanned surgery. A wide excision in such cases would leave
no cutaneous tissue or a very thin layer that is prone to necrotize. Classically an amputation is indicated in this situation. In our series, we chose intercalary resection of the limb, including circumferential skin in some cases. This procedure reduces concerns of necrosis by reconstructing the region of concern at initial surgery. Furthermore, the unaffected distal part was saved with a normal limb length and an adequate surgical margin was achieved. The limitations of this study include the small and heterogeneous study population and short follow-up time. In our hospital, massive intercalary resection and reconstruction of the lower limb accounts for 5.6% of surgeries (excluding biopsies) for primary extremity malignancy. Future studies with more patients and longer follow-up are required to ensure the oncologic and functional effectiveness of our procedure, although the indication applies to only a small proportion of cases with extremity malignancy. In conclusion, massive intercalary resection and reconstruction of the lower limb produced reasonable short-term local outcomes from an oncologic perspective. The unaffected
Fig. 2 Illustrative case of a 6-year-old patient with osteosarcoma of the right femoral shaft. (A) Preoperative MR scan. The case was thought to be an indication for hindquarter amputation. (B) An “en bloc” resection of the femoral lesion was performed, including the femoral nerve and a full segment of quadriceps muscle, but saving the femoral artery and vein, the sciatic nerve, and a small proportion of hamstring muscles. (C) The femur was reconstructed with a composite using the MUTARS for the proximal femur and a structural allograft for the distal femur. The knee extensor was reconstructed with a neuromuscular free flap with anastomosis of the thoracodorsal nerve and the muscular branch of the femoral nerve. Finally, coverage with a myocutaneous free flap was performed, and the thoracodorsal artery and femoral artery were anastomosed in an end-to-side manner. (D) Postoperative X-ray. At 9 months after surgery, he could actively extend the knee with an extension lag of 15 degrees and walk with a crutch outdoors. Journal of Reconstructive Microsurgery
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15 Wright J, Doan T, McBride R, Jacobson J, Hershman D. Variability in
16
17
Conflict of Interest None. 18
Funding None. 19
Financial Disclosure None. 20
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Vol. 30
No. 4/2014
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distal part and the length of involved limb can be preserved by the procedure even in cases with a locally advanced or complicated malignancy. Because of patient preference and fair functional outcomes, we believe the procedure should be viewed as a useful alternative for the treatment of malignant tumors otherwise indicated for amputation.
Shin et al.
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