malignant bone tumors - sparing vs amp

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Vol 11, No 1, January/February 2003 25

Primary malignant bone tumors arerare lesions, with fewer than 3,000new cases per year in the UnitedStates. Before the 1970s, manage-ment routinely consisted of trans-bone amputations or disarticula-tions, with dismal survival rates(10% to 20%). With the develop-ment of more effective chemothera-peutic agents and treatment proto-

cols in the 1970s and 1980s, survivalrates improved, which allowed thefocus of management to shift tolimb preservation.1 Computedtomography and magnetic reso-nance imaging (MRI) allow the pre-cise visualization of the anatomiclocation of a tumor and its relationto surrounding structures. Preop-erative planning has been advancedthrough the use of these modalities,fostering better patient selection for

specific treatment strategies andlowering the morbidity rates ofbiopsy and subsequent resection.2,3

Currently, 80% to 85% of patientswith primary malignant bonetumors involving the extremities(eg, osteosarcoma, Ewings sarco-ma, and chondrosarcoma) can betreated safely with wide resectionand limb preservation. Multi-

modality therapy has increasedlong-term survival rates of patientswith chemotherapy-sensitive tumorsto 60% to 70%.1,4 There are a num-ber of options for skeletal recon-struction after bone tumor resection,and it is important to compare theclinical and especially functionaloutcomes based on type of recon-struction, location of the tumor, andlimb-sparing versus ablative sur-gery.

Limb-Sparing SurgeryPrinciples and Guidelines

There are four basic principles orgoals of limb-sparing procedures:

(1) Local recurrence should be nogreater and survival no worse thanwith amputation. (2) The proce-dure, or treatment of its complica-tions, should not delay adjuvanttherapy. (3) Reconstruction shouldbe enduring and not associated witha large number of local complica-tions requiring secondary proce-dures and frequent hospitalizations.(4) Function of the limb shouldapproach that obtained by amputa-

tion, although body image, patientpreference, and lifestyle may influ-ence the decision.5

Before consideration of limbpreservation, the patient needs to beappropriately staged and assessedthrough a multidisciplinary ap-proach. Some elements of the dis-ease may warrant concern, includ-

Dr. DiCaprio is Resident, Department ofOrthopaedics and Rehabilitation, Yale Univer-sity School of Medicine, New Haven, CT. Dr.Friedlaender is Wayne O. Southwick Professor

and Chair, Department of Orthopaedics andRehabilitation, Yale University School of

Medicine.

Reprint requests: Dr. DiCaprio, PO Box208071, New Haven, CT 06520-8071.

Copyright 2003 by the American Academy ofOrthopaedic Surgeons.

Abstract

Amputation, once the mainstay of treatment of malignant bone tumors, nowis used selectively and infrequently. Most patients are candidates for limb-sparing procedures because of effective chemotherapeutic agents and regi-mens, improved imaging modalities, and advances in reconstructivesurgery. Patient age as well as tumor location and extent of disease help

define the most appropriate surgical alternatives. Options for skeletal re-construction include modular endoprostheses, osteoarticular or bulk allo-

grafts, allograft-prosthetic composites, vascularized bone grafts, arthrodesis,expandable prostheses, rotationplasty, and limb-lengthening techniques.Two key factors must be considered: survival rates should be no worse thanthose associated with amputation, and the reconstructed limb must providesatisfactory function. Functional outcome studies comparing limb-sparing

procedures and amputation have inherent limitations, including the inabilityto randomize treatment and the subjective nature of important outcomemeasures.

J Am Acad Orthop Surg 2003;11:25-37

Malignant Bone Tumors:

Limb Sparing Versus Amputation

Matthew R. DiCaprio, MD, and Gary E. Friedlaender, MD


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ing relative contraindications tosuch procedures (Table 1). Multi-drug neoadjuvant chemotherapy,popularized first for patients withosteosarcoma by Rosen in the late1970s, is usually initiated as appro-

priate after histologic diagnosis andstaging. Chemotherapy helps con-trol systemic disease by attackingmicrometastases, dramaticallyincreasing overall survival rates.1

Neoadjuvant therapy also steril-izes the reactive zone around thetumor by destroying microscopicdisease at the periphery of the pri-mary lesion, thus facilitating resec-tion. Additionally, in some patientswith a relative contraindication to

limb salvage, such as a pathologicfracture in the upper extremity, theuse of chemotherapy with a favor-able response may allow limb sal-vage to be considered. However,not all malignant bone tumors (andespecially chondrosarcomas) have aviable and effective chemotherapyregimen.

When appropriate, after 8 to 12weeks of preoperative neoadjuvantchemotherapy, wide tumor resec-

tion is performed to establish localtumor control. Achieving tumor-free resection margins is of para-mount importance and remains theprimary goal in surgical oncology.Most resections are performedthrough an extensile longitudinalincision, permitting access to themajor neurovascular bundle, withcomplete removal of all biopsytracts. By definition, a wide resec-tion will include a cuff of normal tis-

sue surrounding the resected speci-men. Skeletal defects are large,averaging 15 to 20 cm, reflecting thesize of these tumors and the needfor negative margins. After recon-struction, muscle transfers may benecessary to provide adequate motorfunction. Finally, adequate, healthysoft-tissue coverage is essential toprevent early wound complicationsand subsequent infection. Coveragemay require complex local flaps or

even free tissue grafts. When in-dicated on the basis of histologic

diagnosis, adjuvant chemotherapyconsisting of multiple agents forsynergistic activity is continued for6 to 12 months after wide tumorresection.

After negative tumor margins areobtained, there is often a large skele-tal defect requiring reconstruction.Several options are available.6 Pa-tient age, tumor location, and extentof disease narrow the list of appro-priate surgical alternatives.

Modular Endoprostheses

Indications and Advantages

Currently available metallic pros-thetic systems offer a lightweight,strong, inert means for skeletalreconstruction. Modularity of pros-thetic design allows intraoperativeflexibility based on the final amountof tissue resected. A rigorous reha-

bilitation program can be initiatedimmediately after implantation(usually done with bone cement),allowing early joint range of motionand weight bearing. Prostheticreconstruction carries a lower risk ofdeep infection than do allografts,and nonunion is not a concernbecause there are no osteosynthesissites. Endoprosthetic use alsoavoids the risk of disease transmis-sion and immune responses that

exists with allograft reconstruction.Longevity, complications, and func-

tional outcome vary by anatomicsite, type of prosthesis, and fixationtechnique.

Complications and

Clinical Results

Early complications associatedwith the extensive nature of mostmusculoskeletal oncology proce-dures include wound necrosis/dehiscence, infection, thromboem-bolic disease, neurapraxia, and joint

instability. Meticulous surgical tech-nique and attention to soft-tissuehandling and reconstruction cansignificantly decrease the frequencyof these complications. Late compli-cations include aseptic loosening,infection, joint or prosthetic insta-bility, fatigue fracture of the pros-thesis, and wear or dissociation ofmodular components. Late infec-tion remains the most serious prob-lem because most prosthesis-related

complications can be successfullytreated with revision surgery. Con-cerns include the suboptimal attach-ment of soft tissues to the metalliccomponents.

In an attempt to determine pros-thesis and extremity survivorship,Horowitz et al7 reviewed their expe-rience with 93 prosthetic reconstruc-tions over 8 years: 16 proximalfemur, 61 distal femur, and 16 proxi-mal tibia. Minimum follow-up was

Malignant Bone Tumors: Limb Sparing Versus Amputation

Journal of the American Academy of Orthopaedic Surgeons26

Table 1Relative Contraindications to Limb-Sparing Procedures

Major neurovascular structures encased by tumor when vascular bypassis not feasible

Pathologic fracture with hematoma violating compartment boundaryInappropriately performed biopsy or biopsy-site complications

Severe infection in the surgical field

Immature skeletal age with predicted leg-length discrepancy >8 cm

Extensive muscle or soft-tissue involvement

Poor response to preoperative chemotherapy


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24 months (mean, 80 months).Prosthesis survival at 5 years was88%, 59%, and 54% for proximalfemur, distal femur, and proximaltibia reconstructions, respectively.The overall event-free prosthesis

survival was 63% at 5 years and 36%at 10 years. Aseptic loosening wascause for failure in approximately20% at 5 years and 30% at 10 years.Limb survival for the entire groupwas 87% at 5 years and 81% at 10years. Patients with lesions of theproximal tibia had the longest sur-vival rate, with 93% alive at 10 years.(Histologies included 11 osteosarco-mas, 4 malignant fibrous histiocy-tomas, and 1 chondrosarcoma.) The

group as a whole had a survival rateof 72% at 10 years and was com-posed of a variety of histologies,including 65 osteosarcomas, 13chondrosarcomas, 10 malignantfibrous histiocytomas, 3 Ewings sar-comas, and 2 liposarcomas.

Aseptic loosening is the primarylong-term concern with this methodof reconstruction for tumors aroundthe knee. Whereas allografts success-fully stabilize after 3 to 5 years, pros-

theses begin to exhibit their inherentbiomechanical limitations after 10years. For the current rotating-hingeknee design, reported follow-up islimited to approximately 10 years.Malawer and Chou8 in 1995 showedan 83% survival of prostheses at 5years and 67% at 10 years. Of 52patients who survived and wereavailable at 3-year follow-up, only 10were available at the 10-year follow-up. They had a revision rate of 15%,

infection rate of 13%, amputation rateof 11%, and local recurrence rate of6%. Overall, 44% of patients had atleast one complication. These sur-vivorship data are limited because ofthe small number of patients but mayrepresent an improvement from thesimple-hinge, custom-made prosthe-ses, for which 5-year survival of 80%drops to 53% at 20 years.7,9-11

Of 1,001 patients treated withcemented, custom-made endopros-

theses, aseptic loosening was theprincipal mode of failure among the210 requiring revision.10 Seventy-four revisions (35.2% of those re-vised) were done for aseptic loosen-ing. At 10 years, the rates of aseptic

loosening were 6.2%, 32.6%, and42% for the proximal femur, distalfemur, and proximal tibia, respec-tively. The poorest prognosis forprosthesis survival without asepticloosening was in young patients(


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because of tenuous soft-tissue cover-age and unreliable extensor mecha-nism reconstruction. Survival ratesvary from 45% to 74% at 5 years and45% to 50% at 10 years.9 Malawerand Chou8 found proximal tibial

replacements to have the highestcomplication and revision rateand worst Musculoskeletal TumorSociety (MSTS) functional scores(Table 2) for any region reconstruct-ed. Wound problems and subse-quent infections have decreasedwith the routine use of flaps butremain frequent problems for recon-structions in this anatomic site.Grimer et al17 reported an initialinfection rate of 36% that was re-

duced to 12% by the use of a medialgastrocnemius flap. Local recur-rence was observed in 12.6% ofpatients and was associated withpoor response to chemotherapy andclose margins of excision. They alsofound that 70% of patients at 10years required further surgical pro-cedures and reported a 25% risk ofamputation at 10 years.17

Many designs and techniques areavailable for proximal humeral

replacements. The technique ofMalawer and Chou,8 includingimplantation of a large-segmentprosthesis stabilized by static recon-struction with Dacron tape anddynamic reconstruction by muscle

transfers, has yielded the best re-ported results of any site of pros-thetic replacement, with an averageMSTS functional score of 86.7%(26/30) and no cases of instability.However, there is a wide range of

results reported for this site, with in-stability the primary reason for poorfunctional outcome.7,9,11 OConnoret al18 reported on 11 patients whounderwent proximal humeral pros-thetic reconstruction. Two demon-

strated evidence of stress shielding,six had signs of instability, one hada deep infection, and two had ce-ramic prosthesis loosening or frac-ture. Four of the 11 patients wenton to secondary arthrodesis. Those



A B C D

Figure 1 A 16-year-old boy presented with knee pain of 7 months duration and a history of bilateral retinoblastoma in infancy.A, Anteroposterior radiograph of the knee shows an aggressive, eccentric, osteoblastic distal femoral metaphyseal lesion with extensive

periosteal reaction (Codmans triangle, arrow). Coronal (B) and sagittal (C) T2-weighted MRI scans demonstrate the heterogeneouslesion, periosteal reaction (white arrow), and soft-tissue extension (black arrow). Open biopsy confirmed the diagnosis of high-gradeosteosarcoma. Treatment included preoperative chemotherapy, wide tumor resection, skeletal reconstruction, and postoperativechemotherapy. D, Anteroposterior radiograph after distal femoral reconstruction with a modular rotating-hinge knee prosthesis.

Table 2Musculoskeletal Tumor Society Functional Evaluation16

Lower Extremity Data* Upper Extremity Data*

Pain PainFunction FunctionEmotional acceptance Emotional acceptanceUse of supports Hand positioningWalking ability Manual dexterityGait Lifting ability

* Within each category, each of the six factors is graded 0 to 5. The higher the number,the greater the improvement in outcome. The sum total for the six categories canequal a Maximum Extremity Score of 30 points (5 6). Outcome is reported as a per-centage of the Maximum Extremity Score.


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not converted to arthrodesis weresatisfied regarding pain, emotionalacceptance, and manual dexterityand were dissatisfied regardingfunction, positioning of the hand,and lifting ability.

Osteoarticular or BulkAllografts

Indications and Advantages

Frozen allografts have been usedlonger than any other tumor recon-struction option. Allografts arefavored by some for their potentialfor longevity because they functionas a biologic reconstruction. Incor-

poration of the allograft by the hostis a slow and incomplete process.Osteoarticular allografts permit theuninvolved portion of the joint to bepreserved; this approach allows thestrongest means of soft-tissue orperiarticular ligament reconstruc-tion (Fig. 2). Although associatedwith more early complications thanare endoprostheses, allograft recon-structions stabilize after 3 to 5 yearsand therefore do better in long-term

follow-up studies. Mankin et al19

found that, after 3 years, approxi-mately 75% of grafts are retained bypatients and remain successful formore than 20 years. Seventy per-cent to 80% of patients obtain agood or excellent functional resultafter allograft reconstruction,although this varies with type ofgraft, anatomic site, and stage ofdisease.19 The unpredictable earlyoutcomes with allografts and the

frequent need for multiple proce-dures to obtain a successful endresult have led many orthopaediconcologists to favor modern endo-prostheses. With their potential forlong-term stability, however, allo-grafts play a key role in youngerpatients (


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allograft and those who did not.There was a trend toward a higherincidence of nonunion in patientswith fractures, but it was not statisti-cally significant. The mean time tofracture was 28.6 months after the

index surgery, with more than 70%of fractures occurring within 3 years.The results in this population aftertreatment approached those of pa-tients who had never had a fracture.The mean time to union was 7.4months (range, 4 to 14 months), withall but four fractures treated with anoperation and most involving autog-enous bone grafting. Weight bear-ing was restricted until radiographicunion. The 43 patients underwent a

total of 59 operations. The authorsconcluded that 9.3% of allograftshaft fractures (4/43) may heal withimmobilization, but many requiretreatment with internal fixation andbone grafting. Several attempts maybe necessary, and sometimes ex-change of the allograft or conversionto a metallic endoprosthesis is neces-sary.

Berrey et al21 classified the frac-tures into three patterns. Type I

fractures (2/43) were seen soon aftersurgery, with almost complete dis-solution of the graft. These werethought to be secondary to an im-mune reaction to the allograft. TypeII fractures (22/43) were through theshaft of the allograft, with a meantime to fracture of 27.6 months.Type III fractures (19/43) occurredat the articular surface of osteoartic-ular allografts at a mean of 31.6months from surgery. These are

best treated with a standard resur-facing total knee arthroplasty, whenfeasible.

In a larger series of allograft re-constructions,22 fractures occurredat a rate of 17.7% (185/1,046). Meantime to fracture was 3.2 years.There were 8 Berrey type I fractures,114 type II, and 63 type III. Sixty-one fractures (33%) involved a screwhole at the end of the plate. Neitheradjuvant chemotherapy nor radia-

tion influenced the rate of allograftfracture.

Nonunion is another commoncomplication in the postoperativecourse of allograft reconstructions.All allografts have at least one os-

teosynthesis site that, until healed,limits the amount of weight bearingpermitted through the reconstructedlimb. The location of the osteosyn-thesis affects the healing potential.Diaphyseal-to-diaphyseal osteosyn-thesis sites have a higher risk ofdelayed union or nonunion than dometaphyseal-to-metaphyseal sites.Supplemental autograft and strongerinternal fixation are recommendedat osteosynthesis sites in an effort to

decrease the rate of nonunion. Inthe future, bone morphogenetic pro-teins may play a similar role whenallograft reconstruction is per-formed. Hornicek et al23 evaluatedfactors affecting nonunion of theallograft-host junction. Of 945 pa-tients, 163 (17.3%) had a nonunion.Those receiving chemotherapy hadtwice the rate of nonunion. Twohundred sixty-nine additional sur-geries were performed on these 163

patients. In 114 patients, treatmentled to successful union. The per-centage of failure increased as thenumber of surgical procedures in-creased. Despite treatment, 49 pa-tients failed to demonstrate union ofthe osteosynthesis site.

In comparing different types ofallografts, intercalary allografts havebetter cl inical outcomes than doosteoarticular allografts, allograft-prosthetic composites, and allografts

used for arthrodesis6,19,24 (Fig. 3).Ortiz-Cruz et al24 reviewed 104 in-tercalary allografts done over an 18-year period (median follow-up, 5.6years). Eighty-four percent (87/104)were considered successful, withretention of allograft and normalextremity function. Infection, frac-ture, stage of disease, and adjuvanttherapy all had adverse effects ongraft survival. Fifteen reconstruc-tions failed, most within 3 to 4 years.

Four were salvaged with a secondallograft, three by another recon-struction technique; eight requiredamputation (two for local recur-rence). With two osteosynthesissites, nonunions might be expected

to be a common concern with inter-calary allografts. Thirty-one of the104 allografts (30%) failed to unite atone or both junctions within 1 year,but only seven remained ununited(and were considered failures) afteradditional surgical intervention.Eighty-one additional surgical pro-cedures were needed to achieve sat-isfactory function in 92% of thesepatients. Different modes of internalfixation were used; plate fixation

spanning both osteosynthesis siteswas found to be superior to the useof two shorter plates at either end ofthe allograft.

Mankin et al19 found similar re-sults in their review of 718 allografttransplantations (mean follow-up,78 months). Intercalary allograftsyielded the greatest satisfaction,with excellent or good outcomes in84% of cases. Osteoarticular allo-grafts, allograft-prosthetic compos-

ites, and allograft arthrodeses hadexcellent and good outcome rates of73%, 77%, and 54%, respectively,although they often required addi-tional surgical procedures to achievethese outcomes.

Regional Considerations

Most allograft reconstructionsare for the femur, and the results oflarge studies primarily reflect theoutcome for this region. Hornicek

et al25 reviewed the largest series todate of proximal tibial osteoarticularallografts, consisting of 38 recon-structions (38 patients) in 15 years.Fifty-five percent of the patientsexperienced one or more complica-tions, which were managed withmultiple subsequent procedures.Three amputations were done fordeep infections. About one third ofthe patients required removal of theoriginal allograft and reconstruction




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with a new allograft or conversion

to metallic prosthesis. Ultimately,66% had a good or excellent func-tional result. The study also com-pared outcomes and complicationsbetween the subset of patients treat-ed with chemotherapy, radiationtherapy, or both to those withoutadjuvant therapy. The only signifi-cant (P < 0.05) difference observedbetween the groups was the higherincidence of fracture in the patientstreated with chemotherapy.

Proximal humeral osteoarticularallografts are an attractive option be-cause of their potential for soft-tissuereconstruction, healing, and func-tion. OConnor et al18 reported oneight patients treated with this tech-nique, a subset of 57 patients whounderwent limb-sparing tumor re-sections and various forms of recon-struction. There was no nonunionor cases of instability, but half of thepatients experienced subchondral

fractures and collapse of the articu-

lar surface. Three of these four pa-tients were asymptomatic; the otherwas treated with conversion to aprosthesis. Functional rating aver-aged 71% by the MSTS system, withpatients least satisfied with functionand positioning of the hand. Com-pared with endoprosthetic recon-struction, osteoarticular allograftsresulted in superior function afterintra-articular resection of the proxi-mal humerus. Shoulder arthrodesis

using an intercalary allograft com-bined with plate fixation and vascu-larized fibular grafting is an excel-lent method of reconstruction afterextra-articular resection of the proxi-mal humerus.

Getty and Peabody26 reportedsimilar results in 16 patients whounderwent osteoarticular allograftreconstruction after intra-articularresection of the proximal humerus.At a mean follow-up of 47 months,

the mean MSTS functional evalua-

tion score was 70%. Deteriorationwas noted to continue with timefrom surgery. The authors havestopped doing the procedurebecause of the unacceptable ratesof epiphyseal fragmentation (4/16),instability (11/16), fracture (4/16),and infection (1/16).

Allograft-Prosthetic Composites

Clearly there are benefits andinherent drawbacks to either allo-

graft or endoprosthetic reconstruc-tion. By combining the two meth-ods or using an allograft-prostheticcomposite, the surgeon can tailorthe procedure to help diminish theinherent risks encountered wheneither reconstruction is used alone.The composite helps restore asmuch bone stock as possible andoffers joint stability that is oftendifficult to obtain with osteoarticu-lar allograft reconstruction. By



Figure 3 A 21-year-old man presented with thigh pain. Anteroposterior (A) and lateral (B) radiographs of the femur show a permeativediaphyseal lesion with cortical erosion (white arrows). C, Coronal T2-weighted MRI scan demonstrates the diaphyseal lesion (blackarrow). Open biopsy confirmed Ewings sarcoma. D, Postoperative anteroposterior radiograph of the femur after wide tumor resectionand intercalary allograft reconstruction stabilized with a statically locked intramedullary nail.

A B C D


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resurfacing the allograft bone withan implant, cartilage degradationis no longer a potential problem 5to 10 years after reconstruction(Fig. 4).

Gitelis and Piasecki14 performed

11 hip and 10 knee reconstructionsand 1 elbow reconstruction in 22 pa-tients (mean follow-up, 45 months).Mean MSTS functional score was94.3%. Five patients had a non-union, four of which healed afterbone grafting; one was converted toa megaprosthesis. There were nodislocations. Graft resorption didnot occur in this small number of pa-tients, and no revisions were donefor implant loosening.

Techniques With SpecialIndications

Vascularized Bone Grafts

Vascularized bone grafts can betaken from the iliac crest, rib, scapula,or fibula. Of these options, only vas-cularized fibular grafts are suited forthe large skeletal defects left afterwide resection of a malignant bone

tumor. Compared with allografts,vascularized autografts offer a morerapid incorporation, stronger initialconstruct secondary to graft hyper-trophy, and absence of immunologicproblems. Vascular grafts changenot the pattern of bony repair butrather the rate of repair. Final matu-ration and hypertrophy of graftsis consistent with Wolffs law.29

External fixation is preferable toplate fixation because it maximizes

these stresses and allows for greaterhypertrophy and ultimate strengthof the graft. Ideally suited for chil-dren and young adults, this methodof biologic reconstruction has thepotential to be enduring withoutneed for revision surgery later in life.Vascularized autografts also are usedwith the poorly vascularized tumorbed commonly found in previouslyirradiated tissue and when a delay inosteosynthesis healing is anticipated

secondary to adjuvant therapy withradiation, chemotherapy, or both.The main disadvantages of vascular-ized autograft are the increasedsurgery time, surgical site morbidity,

and size limitations.Few published reports focus on

vascularized autograft in tumorreconstruction, and long-term out-come data are lacking.30 Hsu et al31

reviewed a consecutive series of 30patients who underwent skeletalreconstruction by vascularized fibu-lar transfer after resection of primarybone tumors. Mean follow-up was36 months (range, 24 to 85 months),with union achieved in 90% (27/30)

at an average of 7.6 months. Themean fibular graft length was 18.9cm (range, 10 to 30 cm). Functionalresults were evaluated in 24 patients,with 9 excellent, 7 good, 6 fair, and 2poor results. When used for inter-calary grafts (14 patients), the func-tional results were better than thoseseen with arthrodesis procedures (10patients). There was a high compli-cation rate (50%), but many weremanaged nonsurgically and resolved

without greatly affecting the finaloutcome. Complications includedthree nonunions, three deep infec-tions, three stress fractures, two localrecurrences, and an assortment of

soft-tissue complications.

Arthrodesis

Arthrodesis creates a stable,painless, durable limb. Indicationsfor arthrodesis are extra-articularjoint resection or extensive muscleresection with lack of remainingmuscle to power the joint, or whenthe desire for joint stability is para-mount. The two most common re-gions for this technique are the knee

and shoulder.Knee arthrodesis can be accom-

plished with allografts, nonvascu-larized autografts, vascularizedrotational fibular grafts, external fix-ation with bone transport, or somecombination of these techniques.Fixation is achieved with eithercompression plating or intramedul-lary nailing. Intramedullary fixa-tion is favored for arthrodesis be-cause of a decreased rate of graft



Figure 4 A 14-year-old boy presented with knee pain of 3 months duration and anenlarging mass. A, Anteroposterior radiograph of the knee demonstrates an eccentric lyticproximal tibial metaphyseal lesion with cortical destruction and soft-tissue extension(arrow). Biopsy confirmed osteosarcoma. Anteroposterior (B) and lateral (C) radiographsdemonstrate skeletal reconstruction with a rotating-hinge knee prosthesis and proximaltibial allograft-prosthetic composite stabilized with a dynamic compression plate.

A B C


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fracture and nonunions. The knee isaligned in 10 to 15 of flexion and0 to 5 of valgus. In the skeletallymature individual, the limb is short-ened 1 to 2 cm to allow for footclearance during the gait cycle. In

skeletally immature patients, thelimb may be lengthened with thegrafting technique; the expectationis that the contralateral normal limbwill continue to grow, with theresult that limb length at skeletalmaturity on the operated side willbe equal or slightly shorter. Theprocedure is associated with a highrate of complications (approximate-ly 50%), including all of the inherentrisks of allograft reconstruction.

Despite this complication rate, how-ever, most patients achieve success-ful union and have a durable, func-tional limb.32-34

The shoulder joint is challengingto reconstruct, given the extremerange of motion and lack of inherentstatic stability. The few publishedreports that discuss shoulderarthrodesis in tumor reconstructionhave small numbers of patients (5 to10). Notable rates of infection, frac-

ture, and nonunion exist, as withknee arthrodesis.32 Most patientsobtain stable fusion, allowing satis-factory function of the upper ex-tremity.18,32

Wolf et al33 reviewed the long-term results in 73 patients who,from 1967 to 1985, underwent resec-tion arthrodesis of the knee withautogenous grafts. Forty patientsfollowed for more than 10 yearsformed the basis of the evaluation.

Intramedullary rods were used tostabilize hemicortical femoral or tib-ial allografts and nonvascularizedautogenous fibular grafts to thenative femur and tibia. A high inci-dence of complications (52%) wasevident, yet most patients eventual-ly achieved a successful outcomewith preservation of the limb, and86% were independent ambulatorsat long-term follow-up. The mostcommon complications were graft

fatigue fracture (51%), delayedunion (23%), rod migration or rodfracture (25%), peroneal nerve palsy(8%), and infection and woundproblems (23%). Thirty-seven of the40 patients (93%) achieved a solid

reconstruction. Two patientsrequired an above-knee amputation,one for infection and the other forlocal tumor recurrence. One addi-tional patient sustained a commi-nuted traumatic fracture of thefusion and was treated with anallograft arthrodesis. The recon-structions proved to be durable,and patient satisfaction and func-tion remained high for decades,with an average MSTS functional

score of 77%.Weiner et al34 evaluated 39 pa-

tients treated with resection arthrod-esis done with an intercalary allo-graft fixed with an intramedullarynail. In 31 patients, this procedurewas the index reconstruction; ineight, it was done after failure of adifferent type of tumor reconstruc-tion. Proximal and distal osteosyn-thesis sites both healed, and functionwas satisfactory in 32 patients.

Nonunion occurred in seven pa-tients (one junction in six patients,both junctions in one). The non-union was healed in five of theseven treated with bone grafting,repeat internal fixation, or exchangeallografting; two patients went on toabove-knee amputation. An addi-tional patient from the study under-went above-knee amputation forlocal recurrence. Fatigue fracture ofthe allograft occurred in five pa-

tients, all within metaphyseal bone.Overall, the rate of complicationwas lower than that experiencedwith autograft arthrodesis.

Expandable Prostheses

Expandable prostheses weredeveloped in an attempt to over-come anticipated limb-length dis-crepancies in the growing childtreated with limb-sparing surgery.The first approach includes the

Lewis Expandable AdjustableProsthesis (LEAP).35 Expansion ofthe LEAP is achieved by a modifiedJacobs chuck mechanism. Rotationof the outer sleeve of the prosthesisengages the threads on the inner

shaft, thereby increasing the lengthof the prosthetic shaft with each rev-olution. An average lengthening is1.5 to 2 cm; the average overallextension capability of a LEAP is 6to 9 cm. Collapse of the expansionmechanism, observed early in itsuse, has been addressed with the useof spacer rings. Problems with tita-nium debris and fatigue failure ofexpandable prostheses led to thealternative use of modular systems.

These systems use a Morse taperlocking system to connect segmentalparts. Exchange of intercalary seg-ments can be performed as thepatient grows, adding 2 cm to thelength of the segment replaced.

When extremity length discrep-ancy reaches approximately 2 cm ormore, an expansion procedure is in-dicated. The original incision is used,and the pseudocapsule around theprosthesis is excised to prevent

problems with joint stiffness afterlengthening and to relieve tension onthe neurovascular bundle. Eckardtet al36 reported on their 14-year ex-perience with 32 expandable pros-theses. Nineteen of the 32 patients(59%) survived, with a median fol-low-up of 105 months. Sixteen ofthe patients (50%) did not undergoan expansion because of death, am-putation, or short duration of follow-up. The remaining 16 patients un-

derwent 32 expansion procedures,to a maximum of 9 cm, withoutinfection. The average time fromimplantation to the first lengtheningwas 19 months. Most of the length-enings were 1.5 to 2 cm. More than50% of the patients had at leastone complication, most frequentlyaseptic loosening or failure of theprosthesis, collapse of the LEAP, tem-porary nerve palsy, or flexion con-tractures. The average MSTS ratings




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were good to excellent at the knee,fair to good at the hip, and fair aboutthe shoulder. The authors concludedthat, in children and the skeletallyimmature, rehabilitation can beproblematic; early loss of joint mo-

tion and fixed flexion contracturescan occur. With advances in tech-nology, a noninvasive prostheticlenghtening mechanism is nowbeing evaluated clinically.

Rotationplasty

Rotationplasty, another recon-struction option in the skeletally im-mature patient, can be done afterwide resection about the knee whenthe sciatic nerve can be preserved.

The tibia is rotated 180 and fusedto the femur, with the ankle jointplaced at the level of the contralateralknee. The procedure creates a func-tional below-knee amputation; out-come far exceeds that associatedwith above-knee ablation. The mainindication for this technique is in avery young child with an extensivemalignant bone tumor and severalyears of growth remaining. Rotation-plasty also may be used in adults

when soft-tissue coverage is inade-quate after extensive tumor resec-tion. In general, the procedure isassociated with a low incidence ofcomplications and a highly func-tional and durable extremity recon-struction.37 Advantages include themaintenance of growth and a func-tioning knee joint, the ability totailor the procedure to obtain limb-length equality at skeletal maturitywithout further operations, an energy-

efficient gait pattern, and avoidanceof the problems of neuromas andstump breakdown seen with ampu-tations. With the advent of expand-able prostheses in the late 1980s,however, the indications for rota-tionplasty have narrowed. A fullpresurgical discussion is essential sothat the patient and family under-stand the advantages and cosmeticappearance of the reconstructedlimb, thus limiting postoperative

dissatisfaction or psychologicalproblems. Meetings with other pa-tients who have had the procedureare beneficial.

Kotz37 reviewed the results of 40patients treated with rotationplasty

between 1976 and 1988. Thirty werefollowed for more than 3 years. Nopatient developed local recurrence;six died from metastatic disease, andthe remaining 24 were tumor free.All patients were prosthetic ambula-tors without additional supports,and most participated in sports.Functional evaluation revealed 68%excellent, 28.5% good, 3.5% fair, andno poor results according to thesystem of Enneking et al.38 Compli-

cations of rotationplasty includepostoperative vascular occlusion,pseudarthrosis between femur andtibia, nerve palsies, rotational mal-alignment, and diffuse osteopenia inthe distal limb bones. The high levelof function achieved by most pa-tients far outweighs the appearanceof the limb, limiting the psychologi-cal problems associated with rota-tionplasty.37,39 Long-term follow-upstudies have shown the high durabil-

ity of rotationplasty, with continuedexcellent or good results at 8 years.39

Limb Lengthening

The Ilizarov bone transport pro-cedure and other techniques of limblengthening can be used to regainbone length after resection and re-construction. Limb lengthening bydistraction osteogenesis or bonytransport has limited utility afterresection of malignant bone tumors

when used as the primary recon-struction technique. The large os-seous defect is difficult to replaceand requires extended periods oftreatment, which are associatedwith significant complications. Fre-quently, the final functional resultwith this technique is poor.40 Limb-lengthening procedures are bettersuited as adjuncts to other methodsof reconstruction or for smallerdefects.

Limb-Sparing ProceduresCompared WithAmputation

Survival and Local

Recurrence Rates

In comparing limb-sparing proce-dures with amputation, the pointsselected as outcome measures areimportant. The primary goal of anyoncologic procedure is local tumorcontrol to diminish local recurrenceand improve overall survival.Limb-preserving procedures havenot decreased overall survivalrates4,5,11,19,41,42 (Table 3). Local recur-rence associated with limb-sparingresection and reconstruction is

slightly greater than it is after ampu-tation or joint disarticulation, butthis has not been found ultimately toaffect patient survival.4,41 In thelargest series to date comparinglimb-sparing surgery with amputa-tion in 227 patients with osteosarco-ma of the distal femur, Rougraff etal41 found local recurrence in 8 of 73patients treated with limb preserva-tion, in 9 of 115 patients treated withabove-knee amputation, and in none

of 39 patients treated with hip disar-ticulation.

Functional Outcome

The MSTS system for assessingthe function of reconstructive pro-cedures (Table 2) is designed toallow for comparison of results.16

Functional outcome studies com-paring limb preservation andamputation have inherent limi-tations, however, including the

inability to randomize treatmentand the subjective nature of impor-tant outcome measures. Most func-tional outcome measures favornonarthrodesis procedures becauserange of motion is measured. Mostof the studies designed to assessfunctional outcome focus on pa-tients with perigeniculate tumorresections. Good and bad out-comes occur with whatever proce-dure is used, and there is little dif-




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ference in quality-of-life outcomesbetween limb-sparing and amputa-tive surgeries.43

Rougraff et al41 found that theirlimb-preservation group had higherfunctional scores than did the grouptreated with amputation but thatthe limb-preservation patients fre-quently required additional surgicalprocedures to reach peak function.Renard et al42 found functional re-sults to be significantly (P = 0.0001)

better af ter limb-saving surgerycompared with ablative therapy;however, complications were threetimes more common in the limb-saving cohort.

Otis et al44 studied the energycost during gait by measuring oxy-gen consumption in 14 patients withcustom-made knee prostheses and12 patients who had had above-knee amputations and been fittedwith an artificial limb. They con-

cluded that prosthetic reconstruc-tion provides superior functionbecause these patients had a lowerenergy cost during gait. In contrast,the patients studied by Harris et al45

functioned similarly and walkedwith comparable velocity, efficien-cy, and rate of oxygen consumptionwhether they had had an amputa-tion, arthrodesis, or arthroplasty.The patients treated with endopros-theses lived more sedentary lives

and were the most protective of thelimb.

Kawai et al46 evaluated clinicaloutcomes, length of resection, andenergy cost of walking after pros-thetic knee replacement for malig-nant tumors of the distal femur. Themean free-walking velocity was 79%of normal, reflecting a decrease inboth cadence and st ride length .Also, mean energy cost during walk-ing was 35% greater than that of

normal control subjects and correlat-ed with the percentage of femur thathad been resected. Hillmann et al47

reported on 67 patients with malig-nant tumors of the distal femur orproximal tibia who were treated byrotationplasty or endoprostheticreconstruction. Patients with a rota-tionplasty had a mean MSTS func-tional score of 80% (24/30) com-pared with 83.3% (25/30) for patientstreated with an endoprosthesis.

Those with a rotationplasty hadfewer restrictions in their daily activ-ities and required ambulatory assis-tive devices less frequently than didthose reconstructed with a prosthe-sis. Finally, McClenaghan et al48

compared oxygen consumption inpatients treated with above-kneeamputation, arthrodesis, or rota-tionplasty. Patients treated by ro-tationplasty walked the most effi-ciently.

Psychological Factors

Whether limb-sparing surgeryoffers a psychological outcome ad-vantage compared with amputationfor extremity sarcomas has yet to bedemonstrated because no long-term

prospective or comparative studieshave been done.5,49 In a small, retro-spective, one-time psychologicalassessment of patients treated forlower extremity sarcomas, no differ-ences between amputation and limbsparing were found regarding cog-nitive capacity, mood, body image,global physical functioning, globaladjustment to illness and surgery,and lifetime prevalence of psycho-logical disorders before or after

surgery.49 Most patients adjust wellto both the disease and the requiredsurgical treatment if they have nopremorbid psychological disorders.5

Costs

Grimer et al43 demonstrated thatendoprosthetic reconstruction is lessexpensive than amputation, basedon a 1997 cost analysis and 20-yearfollow-up. Their formula takes intoaccount the projected need for revi-

sion surgery, based on rates of asep-tic loosening of 2.5% per year andon rates of other causes of early fail-ure (eg, infection, implant failure) of1.5% per year.

Future Directions

As experience is gained, the abili-ty to amend methods of treatmentto improve outcomes will increase.Endoprostheses continue to beimproved. Long-term results of

modular rotating-hinge knee com-ponents are anticipated to yieldbetter results than those of simple-hinge knee prostheses. A few insti-tutions have had experience with anendoprosthesis that is lengthenednoninvasively by the application ofexternal electromagnetic force, anddevelopment continues. Soft-tissuereattachment and ligament recon-struction are easier to perform be-cause of the use of osteoarticular



Table 3Limb Sparing Versus Amputation for Extremity Bone Sarcomas

Management

Outcome Measure Limb Sparing* Amputation

Local recurrence4,5,11,19,41,42 5% to 10% 5%Survival4,5,11,19,41,42 70% 70%Functional outcome41,42,44-47 Good GoodInitial cost43 High LowLong-term cost43 Less than amputation More than limb-

sparing procedure

* 85% to 90% of patients with extremity bone sarcomas can be managed withlimb-sparing surgery.

Only 10% to 15% of patients require ablative surgery as initial management.


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allografts. With the development oftendon-attachment devices or en-hanced tendon-anchorage devicesfor prostheses, the indications andoutcomes for proximal tibial pros-theses are likely to broaden and im-

prove.3 Better stability around thehip and shoulder after prostheticreconstructions fit with these specialdevices should improve functionalresults in these regions. Extracor-tical bone-bridging fixation willlikely improve the longevity ofprostheses by walling off the pros-thesis-bone interface and addingadditional points of fixation toimprove construct strength.3,9

For reconstructions using allo-

grafts, bone morphogenetic proteinsmay decrease nonunion rates. Abetter understanding of allograftbiology, as well as ei ther closermatching of allografts to recipients

or modulation of immune respons-es, may decrease the presumed con-sequences of allograft reconstruc-tion. Using more vascularized bonegrafts and combining them withallografts may help reduce or

address rates of nonunion and frac-ture. The continued vigilant use ofadequate soft-tissue coverage inreconstruction procedures, whichreduced early postoperative woundcomplications, likely will decreasethe incidence of late deep wounds.

Summary

The surgical management of malig-

nant bone tumors of the extremitiespresents many challenges. Withadvances in chemotherapy, radio-graphic imaging, and reconstructivesurgery, most patients with these

rare tumors now can be offeredlimb-sparing surgery. Osteoarticularallografts, modular prostheses, orcomposites of these two approachesform the basis for most currentreconstruction efforts. However,

amputation still plays an importantrole and offers a standard to whichother approaches must be com-pared. Functional outcome andpatient satisfaction appear to be atleast as good, and probably better,after skeletal reconstruction thanafter amputation. However, the sur-gical treatment regimen associatedwith limb-sparing procedures is alsoassociated with significant complica-tions and requires extensive rehabili-

tation. Outcomes should continueto improve as advances are made insurgical technique, implant design,autogenous bone allograft biology,and postoperative management.



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