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01241398-201401000-0001501241398_2014_34_92_wallace_reconstruction_1miscellaneous-article< 89_0_13_6 >Journal of Pediatric Orthopaedics© 2014 by Lippincott Williams & WilkinsVolume 34(1)January 2014p 92–100Results of Cement versus Bone Graft Reconstruction after Intralesional Curettage of Bone Tumors in the Skeletally Immature Patient[Tumors]Wallace, Matthew T. MD, MBA*; Henshaw, Robert M. MD†*Department of Orthopaedics, The George Washington University†Division of Musculoskeletal Oncology, Washington Cancer Institute, Washington Hospital, Washington, DCFinancial Disclosures: None of the authors received financial support for this study.The authors declare no conflict of interest.Reprints: Matthew T. Wallace, MD, MBA, Department of Orthopaedics, The George Washington University, 2150 Pennsylvania Avenue, NW, Washington, DC 20037. E-mail: .AbstractBackground: Resection of periphyseal tumors in children presents several unique challenges and complications. Injury to the adjacent physis during resection and adjuvant application has been associated with adverse growth-related outcomes including angular deformities and physeal arrest. The appropriate method of reconstructing bone defects after resection is also controversial. To date there is scant literature on the use of polymethylmethacrylate (PMMA) bone cement as a method of reconstruction in children, and few long-term studies exist on the incidence of growth-related complications after reconstruction. The objective of this study is to evaluate the mechanical, oncological, and developmental outcomes of PMMA use in children.Methods: The authors retrospectively reviewed the medical records and radiographs of 36 skeletally immature patients who underwent intralesional resections of locally aggressive bone tumors. These patients were divided into 17 patients who received reconstruction with PMMA cement, and 19 patients who were reconstructed with bone graft. Follow-up clinical and radiographic evaluations performed after skeletal maturity were reviewed to assess the structural durability, local tumor recurrence rates, reoperation rates, and the incidence of postoperative complications such as deformity, adjacent joint arthrosis, growth arrest, pain, and functional limitation.Results: The average patient age at the time of surgery was 11.79 years (range, 6 to 15 y). The average length of patient follow-up was 5.3 years (range, 2 to 11.5 y). There were no statistically significant differences observed in the rates of reoperation, local tumor recurrence, growth-related complications, adjacent joint arthrosis, or postoperative pain between the 2 groups. There were no postoperative fractures in the cement group, compared to 3 fractures in the bone graft group, although this was not statistically significant.Conclusions: PMMA cement as a structural augment after resection may be used in the pediatric population for improving the mechanical stability of bone. Cement use is associated with complication rates of arthrosis, local recurrence, and growth complications comparable to those observed with bone grafting.Level of Evidence: Level III: Retrospective comparison study.Benign periphyseal tumors of bone include several of the most common bony lesions encountered in the pediatric population. Many lesions require little more than observation, but for active or aggressive tumors such as aneurysmal bone cysts (ABCs), chondroblastomas, giant cell tumors, and other benign lesions that threaten the structural stability of the bone, resection is often indicated.1 Intralesional curettage, with or without the use of local adjuvants, has been a favored approach for many years for the resection of many of these locally aggressive bone tumors.2 This method of resection aims to obtain local control of the tumor while simultaneously preserving much of the native skeletal architecture to maximize a patient’s long-term functional outcome.Complications of intralesional curettage are often directly attributed to the method of resection. Postoperative fracture, adjacent joint degeneration, local tumor recurrence, and long-term pain or functional limitation may be a direct consequence of the chosen method of resection.3 The extent of the curetted cavity, the local effects of adjuvant therapies on the surrounding bone, cartilage, and soft tissues, and the manner of reconstruction of the bony defect have all been examined for effect on local control and short-term complication rates. Numerous surgical strategies have been defined, but large-scale comparative studies are lacking. What is even less understood is the effect that these strategies have on complications specific to the skeletally immature population.Pediatric tumors of bone present unique challenges to the treating surgeon. Special considerations must be made in the skeletally immature population, as the optimal method of local tumor control must often be weighed against the long-term functional and developmental outcome of the limb. The dynamic and adaptive nature of the immature skeleton makes contemporary limb-salvage treatment approaches both technically feasible and functionally preferable. However, surgical resection for the management of pediatric bone tumors is associated with several potential complications unique to the pediatric population. The presence of an active physis within the zone of resection and iatrogenic damage to the growth plate has been linked to numerous case reports of growth-related complications and progressive limb deformities.4–6 These complications may not develop for several years after surgery, and may not become clinically significant until skeletal maturity. When present, growth complications may require additional surgical management. Most of the published studies on the curettage of benign tumors of bone have focused on short-term rates of local control and early postoperative complications; exceedingly few studies have reported on the long-term growth complications in skeletally immature patients.Of equal uncertainty is the feasibility of use of polymethylmethacrylate (PMMA) bone cement to reconstruct bony defects left after curettage in the pediatric population. Bone grafting of defects is generally the preferred method of reconstruction in the pediatric skeleton with the potential for rapid graft incorporation and remodeling. In the setting of large defects at risk for fracture, or defects adjacent to articular surfaces, the immediate structural support of PMMA offers distinct advantages over bone graft.7,8 Several authors have reported on the use of cement for reconstruction after intralesional curettage, but few reports include a series of pediatric patients, and several authors who advocate cementation frequently perform planned cement removal at a later date.4 To our knowledge, there are no data in the literature that presents the long-term durability and complication rates associated with cement reconstructions in the skeletally immature population. We aimed to evaluate the incidence of structural failure, local recurrence, and physeal arrest or growth disturbance after reconstruction with bone grafting and cementation techniques after curettage of aggressive bone lesions in children.METHODSInstitutional review board approval was obtained for this study. Using the musculoskeletal tumor database at our institution, we identified all patients between 1999 and 2009 treated for benign locally aggressive and/or low-grade malignant bone lesions, specifically lesions resected by intralesional curettage with or without adjuvant cryotherapy, which is the preferred method of local control at our institution for aggressive tumors including aneurysmal bone cyst, chondroblastoma, and giant cell tumor.Of the 244 cases identified, 110 procedures were performed in patients younger than 16 years of age. Patients were included in the study if they met the following criteria: (1) intralesional resection of a locally active benign bone tumor or low-grade malignancy; (2) structural reconstruction of the resulting cavity with PMMA cement or any type of bone graft material (autograft, allograft, or bone matrix); (3) open adjacent growth plates at the time of resection; and (4) clinical and plain x-ray radiographic follow-up at our institution after skeletal maturity. Patients with closing physes or imminent physeal closure, patients with <1 year of expected growth at the time of resection, patients with segmental allograft or endoprosthetic reconstructions, and patients without follow-up after physeal closure were excluded. A total of 36 patients were ultimately included in the study, divided into a series of 17 cementation patients, and a series of 19 bone graft patients. Figure 1 depicts the inclusion of patients in the cementation and bone graft series.FIGURE 1. Inclusion criteria for cementation and bone grafting patient series. PMMA indicates polymethylmethacrylate.The medical records of included patients were reviewed to document the location of resection, histologic tumor diagnosis, incidence of tumor recurrence, defined as the histologically and/or radiographically confirmed return of disease, the need and indication for reoperation, and subjective reports of loss of function or any pain present at most recent follow-up. Available preoperative and postoperative clinical examinations and plain radiographs for included patients were evaluated to document the occurrence of postoperative growth arrest, angular deformity, loosening, migration of cement or hardware, fracture, other indications of failure in the area of reconstruction, or adjacent joint arthrosis, defined as the presence of joint space narrowing, subchondral sclerosis, or the presence of periarticular erosions or osteophytes. Figures 2A and B demonstrate the radiographs of one of the cementation series patients.FIGURE 2. Preoperative radiograph (A) of a 12.5-year-old patient with an aneurysmal bone cyst of the distal radius reconstructed with polymethylmethacrylate cement, and follow-up radiograph (B) at 2.5-year follow-up.Statistical MethodsComplication rates of reoperation, postoperative deformity, arthrosis, growth arrest, pain, and functional limitation were calculated and compared between the cementation and graft series, and these values were compared to historical reports in the literature. χ2 analysis was performed to compare rates of complication between case and control series. Paired t test analysis was performed to evaluate the correlation between patient histologic diagnosis and the incidence of recurrence or complications, and between patient age at the time of surgery and the incidence of complications.RESULTSTables 1 and 2 illustrate the demographic and complication rates for the PMMA (Table 1) and bone grafted (Table 2) patient series.TABLE 1 Polymethylmethacrylate Patient Series: Patient Demographics and Rates of Postoperative ComplicationsTABLE 2 Bone Grafting Patient Series: Patient Demographics and Rates of Postoperative ComplicationsDemographicsThe average patient age at the time of surgery was 11.79 years (12.4 y for cementation patients, 11.3 y for bone graft patients). Ages ranged from 6.5 to 15 years for the PMMA series, and 6 to 15 years for the bone graft series. The average length of patient follow-up was 5.7 years for cementation patients (range, 2 to 11.5 y), and 5.0 years for graft patients (range, 2 to 8). There were 11 cases of aneurysmal bone cyst, 8 chondroblastomas, 6 unicameral bone cysts (UBCs), 3 osteoblastomas, 2 nonossifying fibromas, and 1 case each of fibromyxoma, fibrous dysplasia, desmoplastic fibroma, giant cell tumor, chondromyxoid fibroma, and adimantinoma. The most commonly involved locations were the distal femur and proximal tibia. There was a comparable sampling of ABCs, chondroblastomas, and osteoblastomas between the 2 series, but a greater number of UBCs and nonossifying fibromas in the bone graft series.Outcomes/ComplicationsThere were 3 local tumor recurrences in the cementation group; 1 reported case each for aneurysmal bone cyst and chondroblastoma, as well as a desmoplastic fibroma of the radius that recurred in the ipsilateral ulna. The aneurysmal bone cyst and chondroblastoma were both treated with repeat curettage, cryotherapy, and cementation. The desmoplastic fibroma was treated with ulnar resection and the creation of a single-bone forearm. There were no cases of a second local recurrence in the PMMA group. By comparison, there were 5 local recurrences in the bone graft series, but it is notable that 4 of these cases were multiply recurrent UBCs. The other recurrence was that of an aneurysmal bone cyst that was treated with repeat curettage and bone grafting without recurrence. Figures 3A to C represent 1 case of a local recurrence of chondroblastoma in the PMMA series that was successfully treated with repeat curettage and cementation.FIGURE 3. Preoperative radiograph (A) of a 14-year-old patient with chondroblastoma of the proximal tibia and an ipsilateral nonossifying fibroma. The patient presented 2 years after initial resection with a local recurrence (B) at the time of physeal closure, which was treated with repeat curettage and cementation. Follow-up radiograph (C) at 2.2-year follow-up.In both series, there were no cases of loosening at the graft site or cement-bone interface. There were no postoperative fractures in the cementation group, compared to 3 postoperative fractures in the bone grafting group. All of these fractures were managed nonoperatively, and all had successfully healed by final follow-up. Five patients required a second operation in the PMMA group (rate 29.4%), compared to 6 reoperations in the grafted group (rate 31.6%). In the cementation series, there were 3 reoperations for local tumor recurrence as noted above, 1 case of a septic ankle joint which developed adjacent to retained hardware 9 years after the index procedure, and 1 contralateral femoral shortening osteotomy that was performed for a 4 cm limb-length discrepancy at skeletal maturity in a patient with a previously resected chondroblastoma. In the bone grafting series, there were 4 reoperations for local tumor recurrence, 1 resection of painful heterotopic ossification of the pelvis, and 1 hemiepiphysiodesis performed for progressive tibia valga due to partial physeal arrest after chondroblastoma resection.There was only 1 clinically significant growth complication in each series. As described above, 1 patient in the PMMA series required femoral shortening for a longitudinal growth deformity, and 1 patient who received bone graft required a physeal arrest procedure for an angular growth deformity. Both procedures were required in patients who had previously undergone resection of a chondroblastoma. Three additional patients had postoperative deformities; the previously described case of the single-bone forearm, a second patient who presented with a gross angular deformity of the arm before resection and cementation of a massive aneurysmal bone cyst of the proximal humerus, and a multiply recurrent UBCs that healed in 20 degrees of valgus angulation. None of these patients developed long-term pain or any functional deficits.Four patients developed clinical and radiographic evidence of arthrosis by skeletal maturity, 3 in the cementation series and 1 patient in the bone graft series. The first case was the aforementioned septic ankle. The second case was a chondroblastoma resected from the talus and reconstructed with cement. Another chondroblastoma patient was observed to have chondromalacia of the tibia and femur during knee arthroscopy performed for a sports-related meniscal tear. The first 2 patients developed slight loss of motion at the ankle joint and subtalar joint, respectively, with complaints of intermittent pain with weight-bearing. The fourth case involves a chondroblastoma of the femoral head that was reconstructed with iliac crest autograft after resection. This patient developed radiographic evidence of joint space narrowing in the hip and subchondral sclerosis of the femoral head, but reported only mild pain with prolonged activity. Two other cement patients complained of persistent pain at long-term follow-up. One was a case of plantar fasciitis after resection of an aneurysmal bone cyst of the calcaneus, and the other, a case of complex regional pain syndrome after resection of an aneurysmal bone cyst of the proximal tibia. These were the only 2 cement patients who required daily medication for postoperative pain. By comparison, 3 other bone graft patients complained of pain at long-term follow-up. The patient who underwent resection of painful heterotopic ossification of the pelvis complained of mild positional pain at rest and pain with pressure over the resection site. One patient required a course of anti-inflammatory treatment for pes bursitis over the site of a resected aneurysmal bone cyst. Finally, 1 patient with a multiply recurrent UBCs still reported pain with heavy weightlifting activities.As demonstrated in Table 3, no correlation was found between complication rates and patient age at the time of surgery (P=0.199), or histologic diagnosis (P=0.423). There were no statistically significant differences between the PMMA and bone graft group in terms of patient age, length of follow-up, or complications rates (Table 3).TABLE 3 Statistical Comparison of Cementation and Bone Graft Reconstruction Patient SeriesDISCUSSIONThe primary objective of this study was to determine the long-term structural survival of cement reconstructions and the complication rates of PMMA use compared to bone grafting in the skeletally immature population. We further examined the incidence of growth-related complications after intralesional curettage of locally aggressive benign bone tumors in patients followed to skeletal maturity. Our results suggest that the use of cement in the reconstruction of bone defects after intralesional curettage in the pediatric population is both a technically feasible manner in which to maintain structural integrity of diseased and iatrogenically weakened bone without significantly increasing the risk of physeal-related complications.PMMA as Structural Bone Void FillerDurable reconstructions of bone voids in children are challenging. Smaller bone lengths and narrower medullary canals, less soft-tissue coverage, and poor patient compliance with immobilization and weight-bearing restrictions demand specific consideration as to the method of reconstruction chosen at the time of resection.9 PMMA cement has the mechanical advantage of providing immediate structural support to the bone, absorbing stress, and protecting areas that are at risk for postoperative fracture, including the metaphyseal-diaphyseal junction and subchondral regions of the epiphysis where many tumors invade.4 Pritsch and colleagues reported a 17% postoperative fracture rate after curettage and cryosurgery in adult tumors about the knee, and identified metaphyseal defect-to-bone width ratios >0.6 and 0.8 on anteroposterior and lateral radiograph projections, tumor resections <4 mm in proximity to the joint line, and more cryosurgery freeze-thaw cycles as significant risk factors for postoperative fractures. It was their recommendation that cement reconstructions should be preferred in higher risk resections.7 In the absence of obvious fracture, mechanical failure or loosening can be suggested by fatigue fracture or migration of the cement or hardware, or macromotion-related radiographic lucency independent of local disease recurrence. In our series, we did not observe any evidence of loosening at the bone-cement interface, nor did we observe any postoperative fractures in the cementation group, compared to 3 postoperative fractures in the bone grafting group, although this observation did not reach statistical significance. As this remains the primary objective of our investigation, future research with larger sample sizes would be useful in better evaluating this relationship and minimizing the risk of statistical β-error.When using PMMA for its structural advantages, several authors routinely recommend a second procedure to remove cement and add bone graft at a later date, usually several months after initial resection. This has been the recommended approach because of the presumed mechanical effect of the cement on subchondral bone and joint cartilage, and the uncertainty of the long-term systemic effects of PMMA, even though formal investigation of the difficulty and complication rates associated with such secondary procedures are not routinely discussed.4,10,11 In a series by Turcotte et al,12 there was no difference in the incidence of cartilage damage and subsequent degenerative arthritis between patients receiving cement and those receiving other types of bone void filler after intralesional curettage. In our study, we did not routinely remove cement after resection, and none of our patients required cement removal for reasons other than local recurrence or infection. We observed 3 patients (rate 17.7%) who developed adjacent joint arthrosis with cementation, and 1 patient (rate 5.3%) with arthrosis after bone grafting. This difference was not statistically significant, and is comparable to the 6% to 10% rate of adjacent joint arthrosis or chondral injury observed in previous studies.13,14 In our cementation series, we observed that joint arthrosis was the only factor related to reported or measured loss of patient function.PMMA as Local AdjuvantAnother theoretical benefit of cementation is the adjuvant thermal effect of the curing process that generates local heat. This temperature has been recorded in excess of 60°C, and observed in experimental studies to extend a few millimeters into surrounding bone.15 This has been theorized to be a contributing factor in the use of PMMA to improve local control and decrease recurrence rates in the treatment of several locally aggressive tumors.16 In several small series of giant cell tumors treated with intralesional curettage and cementation, recurrence rates were improved in patients who received cement as an adjuvant, but larger-scale studies and a meta-analysis of prior studies did not find these improvements to be statistically significant.8,17–19 On the basis of historical data, we did not hypothesize or expect to find evidence in support of an adjuvant effect of PMMA, and the large variety of histologic diagnoses present in our treatment groups makes this evaluation difficult. While more aggressive lesions such as chondroblastoma would be expected to have higher recurrence and complication rates compared to self-limiting lesions such as UBC’s and nonossifying fibromas, we did not find a significant correlation between histologic diagnosis and complication rates. We also did not observe any cases of mechanical failure as a result of local disease recurrence. Furthermore, our study did not suggest any potential adjuvant effect of PMMA in comparing patients who received cement reconstruction to those who received bone graft, as this comparison was not found to be statistically significant. Currently, the only strong consensus in the literature on locally aggressive benign bone tumors indicates that the adequacy of resection at the time of surgery, tumor location, and the biological aggressiveness of the lesion are the most important factors associated with predicting local tumor recurrence.5,10,20 In our series, local recurrences occurred in 2/11 cases of ABCs (rate 18.2%), 1/8 cases of chondroblastoma (rate 12.5%), and in the 1 case of desmoplastic fibroma. This is comparable to the 17% to 37% rate of recurrence in previous studies of ABCs,4,10,21 and the 4% to 20% rate of recurrence in prior studies on chondroblastoma.5,14,22,23 Therefore, our results concur with the literature consensus and cannot support a case for a significant adjuvant effect of PMMA after curettage. Our series does suggest that the use of cement in the pediatric population yields rates of local control of benign aggressive bone tumors that are comparable to those achieved by other methods of reconstruction. Larger treatment groups specific to each histologic diagnosis would be the preferred method to better evaluate this.Several authors have observed in their series that bone cement affords a better radiographic and magnetic resonance assessment of local tumor recurrences during follow-up, particularly in the first 2 years after resection, during which bone voids filled with graft may still be in the process of absorption and remodeling.4,10,23 This is a significant benefit considering most local recurrences for benign aggressive tumors are reported to occur within the first 1.5 to 2.5 years, but may occur anywhere between 7 and 51 months.5,10,24,25 For this reason, many authors recommend cementation of larger lesions or more aggressive tumors, or extending the period of time between initial resection and planned cement removal.4,23 More dedicated investigation is required to determine if the use of cement leads to earlier detection of local recurrences.Growth Complications of Intralesional ResectionThe average age of diagnosis of most benign, locally active, or aggressive tumors of bone is the second to third decades of life, usually either around or just after the age of skeletal maturity. However, historical reports estimate that approximately 6% to 10% of giant cell tumors of bone, 7% to 29% of chondroblastomas, and 14% to 50% of ABCs occur in patients with open physes and significant skeletal growth remaining.4,5,10,23,25 The challenge of resection in these cases is to minimize the risk of tumor recurrence with an adequate oncologic resection, while taking care to protect the adjacent physes from iatrogenic damage. Aggressive curettage, high-speed burring, and the use of chemical or thermal adjuvants during intralesional resection of tumors have been cited as potential causes of postoperative complications such as growth arrest, angular deformity, and limb-length discrepancy.4,5,6 Unfortunately, none of the previous studies had followed patients until the termination of skeletal growth to determine the true incidence of these complications.Several authors have noted that during routine preoperative imaging, giant cell tumors of bone, chondroblastomas, and ABCs all have been observed invading and crossing an open growth plate before surgical resection, which may suggest that growth complications after surgery are not entirely iatrogenic in nature.5,10,25 In patients followed long enough to detect growth complications, studies have demonstrated a 7% to 15% rate of physeal arrest after resection of ABCs, and a 7% to 50% rate of arrest after chondroblastoma resection.5,10,26,27 Growth arrest in these studies was associated with a 7% to 17% incidence of clinically significant limb-length discrepancy necessitating treatment either with shoe modification or surgery.11,23 Growth arrest and growth-related deformity each occurred in 5.9% of our PMMA series patients, and 5.3% of our bone graft series patients. Each case of a growth-related longitudinal or angular deformity in our study occurred in a patient treated for chondroblastoma. The longitudinal deformity due to premature physeal closure after cementation required a contralateral femoral shortening procedure to correct a 4-cm limb-length discrepancy. The progressive tibia valga after bone grafting of a proximal tibial chondroblastoma required corrective hemiepiphysiodesis. Rates of growth complications was not found to be clinically significant in our study, and our reported rates of physeal complications are comparable to that of other reported series of chondroblastomas, and other locally aggressive bone tumors. Our results suggest that the use of cement does not significantly increase the risk of postoperative growth disturbance or deformity. This is still an active area of research, and further investigation is required to specifically examine the effect of cement reconstruction on the rates of physeal arrest in long bones, or whether interposition of cement could possibly prevent or delay the formation of osseous physeal bars after intralesional resection.CONCLUSIONSBenign, locally aggressive tumors of bone are challenging problems in the pediatric population. Injury to the open growth plate at the time of resection can lead to growth-related complications and deformity, and may require subsequent surgery. The use of PMMA bone cement as a structural bone void filler after intralesional resection of active benign bone tumors has not been extensively studied to date. Our experience supports the use of PMMA in the pediatric population to augment the mechanical stability of bone after resection, to potentially reduce the risk of postoperative fracture, and to provide better radiographic monitoring for local tumor recurrence. 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[Context Link] cement; bone tumor; skeletally immature; curettage; polymethylmethacrylate; bone cyst; bone graft; chondroblastoma; growth|01241398-201401000-00015#xpointer(id(R10-15))|11065213||ovftdb|SL0000469420022253311065213P63[CrossRef]|01241398-201401000-00015#xpointer(id(R10-15))|11065404||ovftdb|SL0000469420022253311065404P63[Full Text]|01241398-201401000-00015#xpointer(id(R10-15))|11065405||ovftdb|SL0000469420022253311065405P63[Medline Link]|01241398-201401000-00015#xpointer(id(R15-15))|11065213||ovftdb|SL00003086199329523911065213P68[CrossRef]|01241398-201401000-00015#xpointer(id(R15-15))|11065404||ovftdb|SL00003086199329523911065404P68[Full Text]|01241398-201401000-00015#xpointer(id(R15-15))|11065405||ovftdb|SL00003086199329523911065405P68[Medline Link]|01241398-201401000-00015#xpointer(id(R20-15))|11065213||ovftdb|SL0000280819723040111065213P73[CrossRef]|01241398-201401000-00015#xpointer(id(R20-15))|11065405||ovftdb|SL0000280819723040111065405P73[Medline Link]|01241398-201401000-00015#xpointer(id(R23-15))|11065213||ovftdb|SL0000222820111314511065213P76[CrossRef]|01241398-201401000-00015#xpointer(id(R23-15))|11065404||ovftdb|SL0000222820111314511065404P76[Full Text]|01241398-201401000-00015#xpointer(id(R23-15))|11065405||ovftdb|SL0000222820111314511065405P76[Medline Link]20364261Results of Cement versus Bone Graft Reconstruction after Intralesional Curettage of Bone Tumors in the Skeletally Immature PatientWallace, Matthew T. MD, MBA; Henshaw, Robert M. MDTumors134