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Purpose: In this study, we asked the question of whether non-invasive (NI) extendible bone tumour implants are as reliable and reduce infection when compared with patients who received a minimally invasive (MI) extendible implant., Methods: Forty-two NI extendible bone tumour implants were investigated at a mean follow-up of 22 months (range, 1-87 months) and 63 MI implants at a mean follow-up of 49 months (range, 1-156 months)., Results: Kaplan-Meier analysis showed that the probability of MI implant survival was 58.8% compared with 78.6% in NI patients. No significant difference between these two patient groups was found. Infection was the main reason for failure in the MI implant group where nine (35%) implants were revised. However, only one (11%) NI implant was revised for infection ( p = 0.042). None of the NI implants failed due to aseptic loosening; however, six (23%) MI implants were revised for aseptic loosening of the intramedullary stem. Four (15%) of the failed MI implants were revised due to full extension and five (56%) of failed NI implants were replaced as the implant had been fully extended where the patient still required growth., Conclusion: Where possible, an NI massive prosthesis should be used in this patient group. Our results suggest that MI prostheses should be infrequently used due to the high incidence of infection. Lengthening of NI prostheses is painless, can be carried out in the clinic and is more cost-effective. However, further work is required to increase the amount of growth potential available in these implants.

Background: Aseptic loosening of massive bone tumor implants is a major cause of prosthesis failure. Evidence suggests that an osteointegrated hydroxyapatite (HA)-coated collar would reduce the incidence of aseptic loosening around the cemented intramedullary stem in distal femoral bone tumor prostheses. Because these implants often are used in young patients with a tumor, such treatment might extend the longevity of tumor implants. Questions/purposes We asked whether (1) HA-coated collars were more likely to osteointegrate; (2) HA collars were associated with fewer progressive radiolucent lines around the stem-cement interface; and (3) HA-coated collars were associated with less bone loss at the bone-shoulder implant junction?, Methods: Twenty-two patients were pair-matched to one of two groups--either (1) implants with a HA-coated ingrowth collar (HA Collar Group); or (2) implants without an ingrowth collar (Noncollar Group). Age, sex, and length of followup were similar in both groups. HA-coated collars were developed and used at our institution from 1992 to address the high failure rate attributable to aseptic loosening in patients with massive bone tumor implants. Before this, smooth titanium shafts were used routinely adjacent to bone at the transection site. The minimum followup was 2 years (mean, 7 years; range, 2-12 years). Radiographs obtained throughout the followup period were analyzed and osteointegration at the shaft of the implant quantified. Radiolucent line progression around the cemented stem was semi-quantitatively assessed and cortical bone loss at the bone-shoulder implant junction was measured during the followup period., Results: Comparison of the most recent radiographs showed nine of 11 patients had osteointegrated HA collars, whereas only one patient in the Noncollar Group had osteointegration (p > 0.001). The radiolucent line score quantified around the cemented stem was lower in the HA Collar Group when compared with the Noncollar Group (p = 0.001). Results showed an increase in cortical bone loss at the bone-shoulder implant junction in the Noncollar Group when compared with the HA Collar Group (p < 0.001)., Conclusions: Osteointegration at the implant collar resulted in fewer radiolucent lines adjacent to the intramedullary cemented stem and decreased cortical bone loss immediately adjacent to the transection site. These results suggest that the HA collar may help reduce the risk of aseptic loosening in patients with this type of implant, but longer followup and a larger prospective comparison series are necessary to prove this more definitively.

Background: Several different strategies have been reported for the treatment of chondromyxoid fibromas, all with variable outcomes and high recurrence rates., Methods: We report on 22 consecutive cases of chondromyxoid fibromas treated by intralesional curettage, four of which had adjuvant cementation at our institution between 2003 and 2010. We assessed the functional outcome using the Musculoskeletal Tumour Society (MSTS) scoring system., Results: Nine males and 16 females with a mean age of 36.5 years (range 11 to 73) and a mean follow-up of 60.7 months were included in the study. Local recurrence occurred in two patients (9%) within the first 2 years following the index procedure. This was treated by re-curettage only of the residual defect. Two postoperative complications occurred: a superficial wound infection in one patient and a transient deep peroneal nerve neurapraxia in the other. The mean postoperative MSTS score was 96.7%., Conclusions: Intralesional curettage and cementation is as an effective treatment strategy for chondromyxoid fibromas, providing satisfactory functional results with a low recurrence rate. Careful case selection with stringent clinical and radiographic follow-up is recommended.

Background: The objective of this study was to examine the degree of osteointegration into a hydroxyapatite-coated collar and relate this finding to aseptic loosening in patients with a distal femoral replacement used to treat primary bone cancer. Our hypothesis was that the implant collar would increase osteointegration and reduce the rate of aseptic implant loosening., Methods: Sixty-one patients treated with a primary cemented distal femoral prosthesis between 1992 and 2001 were included in this study. The mean duration of follow-up was 8.5 years (range, two to eighteen years). Extracortical bone growth into the grooved hydroxyapatite-coated collar was quantified radiographically. Histological sections through four hydroxyapatite-coated collars and four implants with no collar, retrieved following amputation due to local recurrence or at autopsy at a mean of 3.5 years (range, 1.4 to 6.1 years) after implantation, were evaluated as well., Results: Five (8%) of the implants were revised because of aseptic loosening, 3% of the implants fractured, and 3% were revised because of infection. Six limbs (10%) required amputation because of local tumor recurrence. On radiographs, osteointegration into the collar was seen to have occurred in 70% of the patients and did not correlate with sex, age, diagnosis, or length of time postoperatively. Histological analysis showed mature lamellar bone within the grooves of the hydroxyapatite-coated collar, and bone was observed in direct contact with the hydroxyapatite coating. Extracortical bone failed to make direct contact with the surface of the implants manufactured without a collar., Conclusions: The use of cemented distal femoral massive bone tumor prostheses with a hydroxyapatite-coated collar located at the shoulder of the implant was followed by a low (8%) rate of revision due to aseptic loosening. The use of hydroxyapatite grooved collars may lead to osteointegration of the implant shoulder (collar) and may reduce the rate of aseptic loosening.

Purpose: The optimal reconstructive method after resection of malignant bone tumours of the proximal ulna is unknown.We report the outcome of endoprosthetic replacement in a young patient population., Methods: This was a retrospective review of four patients[three males and one female; mean age 17.5 (range 11–31)years] who underwent limb salvage with a proximal ulnar endoprosthetic replacement following excision of malignant bone tumour. Mean follow-up was 85 (range 14–194) months., Results: All patients were alive at final follow-up and reported an improvement in pain. One patient required transhumeral amputation for intralesional excision complicating a local recurrence at one month. Two patients developed fixed flexion deformities of the elbow, one of whom required radial-head excision. Mean Musculoskeletal Tumour Society (MSTS)score and Toronto Extremity Salvage Score (TESS) were 27(range 25–28) and 81 (73–88), respectively., Conclusions: Custom-made proximal ulna endoprosthetic replacement following resection of malignant bone tumours in young patients provides a stable reconstruction option with satisfactory function and without apparent compromise in patient survival.

Knee arthrodesis is most commonly performed for failed total knee arthroplasty. Conventional arthrodesis techniques are associated with a high incidence of complications and are unsuitable in cases with extensive bone loss. We report our medium-term results using a custom-made cemented knee arthrodesis prosthesis in 10 patients with a mean follow-up of 56.4 months (range, 15-199 months). The prosthesis was implanted as a 1- or 2-stage procedure for infected revision knee arthroplasty or tumor endoprosthesis in 9 patients and as a primary procedure in 1 patient with angiosarcoma involving the knee extensor mechanism. The average combined femoral and tibial bone deficit was 170 mm (range, 56-220 mm). Implant survivorship was 90%. All patients with retained prosthesis had no evidence of residual infection or loosening and were able to mobilize independently. One prosthesis was revised though retained following a prosthetic fracture, and 1 patient underwent above-knee amputation for uncontrolled infection. We conclude that the Stanmore knee arthrodesis prosthesis provides reliable fusion in an otherwise difficult-to-treat group of patients., (Copyright © 2011 Elsevier Inc. All rights reserved.)

The optimal reconstructive method following segmental resection of malignant tumours in the humeral diaphysis is unknown as there are no prospective long-term studies comparing biologic with endoprosthetic reconstruction. This is a retrospective review of 13 patients who, between 1995 and 2010, had undergone limb salvage at our institution using a custom-made humeral diaphyseal endoprosthetic replacement following excision of malignant bone disease. There were 9 males and 4 females with a mean age of 35 years at the time of surgery (range: 10 to 78). Mean follow-up was 56.8 months (range: 5 to 148). Cumulative patient survival was 75% at 10 years. Implant survival, with removal of the endoprosthesis or part of it for any reason as an end point, was 47% at 10 years. Seven patients required revision (54%). Complications included metastases in four, aseptic loosening in four, peri-prosthetic fracture in two and local recurrence in two. Mean MSTS and TESS scores were 23 (18 to 27) and 67% (52-80) respectively. Custom-made humeral diaphyseal replacement following resection of malignant bone tumours provided functional results superior to amputation, without an obvious compromise in patient survival. There was a relatively high revision rate for aseptic loosening and peri-prosthetic fracture and patients should be counselled about this preoperatively.

Chordoma, the molecular hallmark of which is T (brachyury), is a rare malignant bone tumour with a high risk of local recurrence and a tumour from which metastatic disease is a common late event. Currently, there is no effective drug therapy for treating chordomas, although there is evidence that some patients respond to the empirical use of epidermal growth factor receptor (EGFR) antagonists. The aim of this study was to determine the role of EGFR in the pathogenesis of chordoma. Paraffin-embedded material from 173 chordomas from 160 patients [sacro-coccygeal (n = 94), skull-based (n = 50), and mobile spine (n = 16)] was analysed by immunohistochemistry and revealed total EGFR expression in 69% of cases analysed. Of 147 informative chordomas analysed by FISH, 38% revealed high-level EGFR polysomy, 4% high-level polysomy with focal amplification, 18% low-level polysomy, and 39% disomy. Phospho-receptor tyrosine kinase array membranes showed EGFR activation in the chordoma cell line U-CH1 and all of the three chordomas analysed. Direct sequencing of EGFR (exons 18-21), KRAS, NRAS, HRAS (exons 2, 3), and BRAF (exons 11, 15) using DNA from 62 chordomas failed to reveal mutations. PTEN expression was absent by immunohistochemistry in 19 of 147 (13%) analysed chordomas, only one of which revealed high-level polysomy of EGFR. The EGFR inhibitor tyrphostin (AG 1478) markedly inhibited proliferation of the chordoma cell line U-CH1 in vitro and diminished EGFR phosphorylation in a dose-dependant manner, a finding supported by inhibition of phosphorylated Erk1/2. p-Akt was suppressed to a much lesser degree in these experiments. There was no reduction of T as assessed by western blotting. These data implicate aberrant EGFR signalling in the pathogenesis of chordoma. This study provides a strategy for patient stratification for treatment with EGFR antagonists., (Copyright © 2010 Pathological Society of Great Britain and Ireland. Published by John Wiley & Sons, Ltd.)

We treated three patients with aneurysmal bone cysts and seven with giant cell tumours who presented with a large osteolytic lesion in peri-articular areas of the knee. The patients age ranged from 8 to 49 years (mean 25 years). The average tumour volume was measured at 39.8 cm(3) (range 18 to 65 cm(3)). The tumour cavities were treated with curettage, phenolisation and application of a composite bone graft substitute containing 35% calcium-sulphate hemihydrate and 65% hydroxyapatite granules. All patients were followed up for 4.0 to 5.2 years (mean 4.5 years). Tumour recurrence was noted in two cases. In the remaining patients consolidation of the lesion was considered complete at a mean of 4.5 months (3-6.5 months). During follow-up no deformities developed and no radiological signs of joint degeneration were noted. All patients regained close to normal function, with a mean Musculoskeletal Tumour Society Rating Score of 95.1%. Due to their good osteoconductive abilities, composite synthetic bone graft substitute combining porous hydroxyapatite with calcium-sulphate appears to be an effective alternative to autologous cancellous bone graft in the treatment of large osteolytic lesions in peri-articular areas around the knee joint. They bear major advantages through ubiquitous availability and the avoidance of morbidity associated with iliac crest harvest. Concerns remain as the radio-opaque appearance of the bone graft substitute may potentially delay the detection of tumour recurrence.

Chordomas are rare malignant bone tumours considered to arise from notochordal remnants that persist in the axial skeleton. Although their morphology can resemble that of a carcinoma, chondrosarcoma or malignant melanoma, the axial location and their well-defined immunophenotype, including expression of cytokeratins (CK7/20/8/18/19) and S100, generally allow the diagnosis to be made with confidence once the possibility is considered. In contrast, making a robust diagnosis of an extra-axial chordoma has been difficult in the absence of specific markers for chordomas. We have recently shown in gene expression microarray and immunohistochemistry studies that brachyury, a transcription factor crucial for notochordal development, is a specific and sensitive maker for chordomas. We now present a case of an intracortical tibial tumour, with detailed report of the imaging, and morphological features consistent with a chordoma, where notochordal differentiation was demonstrated with an antibody to brachyury. The tumour cells were also positive for cytokeratins, including CK19, and S100, CEA, EMA and HMBE1, findings which support the diagnosis of chordoma. Brachyury can be employed as a marker of notochordal differentiation and help identify confidently, for the first time, extra-axial bone and soft tissue chordomas, and tumours which may show focal notochordal differentiation.