Abstract
Background
Tumour resection followed by joint reconstruction is a surgical option in the appropriate patient. The evidence for such reconstructive surgery of the elbow joint is limited. The aim of this study is to review the literature to evaluate the outcomes of joint replacement surgery in tumours of the elbow.
Methods
A systematic review of PUBMED and EMBASE databases was conducted. Case series and comparative studies reporting results after total elbow arthroplasty, modular endo-prosthetic replacement and custom prosthesis were eligible for inclusion.
Results
Eleven eligible studies were identified (n = 134). At mean follow-up of 44 months, the overall revision rate was 14% and complication rate was 28%. The mean Mayo Elbow Performance Score was 75, with 56% of patients reporting good or excellent outcomes. The mean post-operative range of motion was 97°.
Discussion
Elbow prosthesis reconstruction after tumour resection can provide good functional outcomes at mid-term follow-up. The complication and revision rates are comparable to other indications for elbow replacement surgery. Further prospective studies are required to compare outcomes between different elbow arthroplasty options after tumour resection.
Introduction
Tumours of the elbow are extremely rare and account for 1% of all tumours presenting to orthopaedic surgeons. 1 The limited soft tissue envelope coupled with the proximity of neurovascular structures makes the surgical management of elbow tumours a lot more challenging than other anatomical regions. These challenges have previously resulted in amputation being an accepted surgical option. 2 However, limb salvage has been reported to have improved functional outcome and cosmesis. 3 The evolution of chemotherapy and radiotherapy have resulted in limb salvage procedures being increasingly considered for treatment of these challenging cases with primary bone and soft tissue tumours. Furthermore, in patients with metastatic disease in the elbow, pathological fracture and limited life expectancy, limb salvage seems more appropriate than ablative surgery.
A multidisciplinary approach must be adopted in the decision-making process between resection with reconstruction or amputation. Key factors considered include tumour size, location, extramedullary extension, metastatic disease, life expectancy and patient factors. Reconstructive options include standard total elbow arthroplasty (TEA), modular endo-prosthetic replacements (EPR), resection arthroplasty, interposition arthroplasty, arthrodesis, elbow osteoarticular allograft reconstruction, allograft-prosthesis composite (APC) arthroplasty and vascularized fibular grafts.1,4,5 Of these options, elbow reconstruction in the form of a prosthesis or APC is more attractive as it has been reported to restore bone defects whilst providing acceptable analgesia, function and complication rates. 6 An inappropriate selection of a standard TEA in patients with large defects after tumour resection could lead to instability with complications like implant loosening and failure. On the other hand, an EPR using a megaprosthesis after significant soft-tissue stripping is unlikely to allow as good a functional outcome as a TEA. 7
It is important to acknowledge that as part of the ‘Get It Right First Time’ approach (GIRFT) to Orthopaedic Practice, Elbow Replacement Surgery has come under particular scrutiny. A robust multidisciplinary approach has been developed by the British Elbow and Shoulder Society (BESS), and from here-on, these management principles will be as important to tumour practice as they are to elbow replacement in non-tumour patients. 8 It is important that an elbow arthroplasty specialist from a designated centre should be involved throughout the management process including surgery.
In addition, each patient should be actively involved in decision making over the surgical reconstruction options. This requires informed consent and discussion of the benefits, potential risks, complications and long-term implications of surgical intervention. However, the current evidence available on patients undergoing elbow replacement surgery for tumours is extremely limited. The aim of this systematic review is to document the functional outcomes, complications and revision rates in this patient group to aid decision making for these complex cases.
Methods
Search strategy
A systematic review was conducted in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. 9 An electronic search was performed on MEDLINE and EMBASE databases for all English language articles with no time filter restriction. The search was performed independently by two authors on 01 September 2020 and repeated on 10 December 2020 to ensure accuracy. The authors also performed searches in the Cochrane library and manual searches in Google using the keywords ‘elbow tumours’, ‘elbow replacement’, ‘elbow arthroplasty’ and ‘endo-prosthetic’. The reference lists of included articles were also screened to identify any articles not initially identified in the preliminary searches. Any discrepancies were resolved through discussion between these two authors, with the senior author resolving any residual differences.
Inclusion and exclusion criteria
Clinical studies published in English were considered for eligibility. Studies could be either case series or comparative studies reporting results after elbow replacement for elbow tumours. Case reports were excluded.
For inclusion, the study must have reported functional outcomes, complications or revision rates. Elbow tumours could be involving either the distal humerus or proximal ulna. The elbow prosthesis used could be TEA, EPR or custom prosthesis, with or without additional use of allografts. Only primary research was considered for review with any abstracts, comments, review articles, case reports and technique articles excluded.
Data extraction
Information was extracted regarding the number of patients in each included study, along with their age, diagnosis, procedure, follow-up, outcomes (range of motion (ROM), post-operative functional scores), complications and revision rates.
Statistical analysis
Descriptive statistics such as mean and range are reported where applicable and percentages were calculated for qualitative data. The heterogeneity and frequent absence of crucial data made comparative statistical analysis difficult. The assessment of methodological quality of the studies was undertaken using the methodological index for non-randomised studies (MINORS) score, where non-comparative studies achieve a maximum score of 16 and comparative studies achieve a maximum score of 24. 10
Results
The search strategy identified 11 studies eligible for inclusion (n = 134): 3 studies on TEA with or without allograft (n = 38),1,11,12 4 studies on EPR (n = 56)7,13–15 and 4 studies on custom replacements (n = 40).16–19 A summary of the included studies is presented in Tables 1 to 3. The demographics according to surgical intervention are provided in Table 4. A flow chart of the search strategy is shown in Figure 1. Table 5 outlines the reasons for article exclusion after full-text review.20–30
PRISMA flowchart. Summary of the included articles for patients receiving TEA. TEA: total elbow arthroplasty; GCT: giant cell tumour; MM: multiple myeloma; APC: allograft-prosthesis composite; ROM: range of motion; MEPS: Mayo Elbow Performance Score; HO: Heterotopic Ossification; E/G: excellent/good; CRPS: Chronic regional pain syndrome. Summary of the included articles for patients receiving modular endo-prosthetic replacement. ABC: aneurysmal bone cyst; GCT: giant cell tumour, UPS: undifferentiated pleomorphic sarcoma, STS: soft tissue sarcoma; MM: multiple myeloma, CCS: clear cell sarcoma; HMRS: Howmedica Modular Resection System; SRS: Segmental Revision System; ROM: range of motion; TESS: Toronto Extremity Salvage Score; MSTS: Musculoskeletal Society Tumour Score; FFD: fixed flexion deformity. Summary of the included articles for patients receiving custom prosthesis. GSB: Gschwend/Scheier/Bähler; GCT: giant cell tumour; MFH: malignant fibrous histiocytoma; HGSSS: high grade soft tissue sarcoma; ROM: range of motion; TESS: Toronto Extremity Salvage Score; MSTS: Musculoskeletal Society Tumour Score; MEPS: Mayo Elbow Performance Score; DASH: Disabilities of the Arm, Shoulder and Hand score. Summary of outcomes from different types of prosthesis. ROM: range of motion; MEPS % E/G: Mayo Elbow Performance Score % of Excellent/Good; MSTS: Musculoskeletal Society Tumour Score; TESS: Toronto Extremity Salvage Score; DASH: Disabilities of the Arm, Shoulder and Hand score; TEA: total elbow arthroplasty. Reason for exclusion of articles after full-text review.
The mean age at surgery for all patients was 56.8 years (21 to 87 years), and the overall mean follow-up duration was 44 months (1 to 372 months), as outlined in Table 4. Seventy-nine patients (59%) receiving elbow arthroplasty were diagnosed with primary tumours, while 55 patients (41%) had a metastatic deposit in the elbow. Primary tumours included aneurysmal bone cyst, giant cell tumour of bone, undifferentiated pleomorphic sarcoma, soft tissue sarcoma, malignant fibrous histiocytoma, high grade soft tissue sarcoma, multiple myeloma and clear cell sarcoma. Metastatic lesions included primary lesions from breast, lung, kidney, colon, uterus, ovary, cervix and larynx.
Functional outcomes
A summary of outcomes from the included studies including ROM (through flexion/extension), functional scores, complications, revision rates and mortality are provided in Tables 1 to 3. These data have been collated in Table 4 into overall means using each type of implant when available.
Range of motion
The average post-operative ROM arc for the three implant types was 97°. The average ROM arc for patients with an EPR was 15° greater than patients with a TEA and 10° greater than those with a custom prosthesis.
Athwal et al. reported the largest series using TEA and demonstrated a statistically significant improvement in ROM from 48° pre-operatively to 92° post-operatively (p < 0.05). 11 Similarly, Capanna et al. presented the largest case series using EPR and reported that two-thirds patients had over 100° arc of motion. 14 The largest such series of custom prosthesis reported by Tang et al. also demonstrated that two-thirds of patients achieved a post-operative ROM of over 100°. 18
Only two studies documented both pre-operative and post-operative ROM, both reporting on TEA and demonstrating statistically significant improvements in post-operative ROM.1,11 Hence, no meaningful statistical change in arc of motion for comparison of different prosthetic types after surgical intervention can be presented. However, it is important to consider that for tumour-related surgery, function is often sacrificed for survival.
Outcome scores
The Mayo Elbow Performance Score (MEPS) was reported in four studies (n = 53). Overall, 70% of the patients had excellent or good scores as demonstrated in Table 4; 80% of patients who received a custom prosthesis reported excellent or good MEPSs, compared to the 66% receiving TEA. There were no MEPSs for patients receiving an EPR. Five studies reported the Musculoskeletal Society Tumour Score (MSTS) (n = 77). The overall mean score for all prosthesis was 80% and was similar after custom prosthesis (79%) and EPR (80%) as illustrated in Table 4. There were no MSTSs for TEA patients. Toronto Extremity Salvage Score (TESS) and Disabilities of the Arm Shoulder and Hand scores were both reported in some studies and the details of these are presented in Tables 1 to 3.
Complications
All included studies reported complications. Overall, there were 38 complications in the 134 patients (28%). The complication rates showed no statistical difference between the groups (p = 0.82); TEA (33%), EPR (29%) and custom prosthesis (25%). Furthermore, there were 17 additional local recurrences during follow-up. The most common overall complications were aseptic prosthesis loosening (11%), nerve injury (10%) and infection (2%). Aseptic prosthesis loosening was due to humeral loosening (7%), ulna loosening (2%) or bushing failure (2%). The other less frequent complications (each at n = 1/134) included heterotopic ossification, united peri-prosthetic fracture, chronic regional pain syndrome, wound healing complication and screw replacement.
The most common complication in the group of patients receiving a TEA was nerve injury (18%). Aseptic loosening occurred in one patient (3%) and there were no reported infections.
The most common complication in the modular EPR group was aseptic prosthesis loosening (16%), followed by nerve injury (7%) and infection (4%). Permanent nerve palsies were only recorded in patients who underwent implantation of endoprosthesis (n = 2, both radial nerve) and they were both in the same study. 14
The most common complication in the custom prosthesis group was aseptic prosthesis loosening (13%), followed by nerve injury (8%) and infection (5%).
Revisions
The overall revision rate for all 134 patients was 14% with a mean follow-up of 44 months. The revision rate for TEA was the lowest at 3% (follow-up 36 months), custom prosthesis was higher at 18% (follow-up 42 months) and modular EPR was the highest at 20% (follow-up 49 months). Despite the apparent difference in revision rates, this was not statistically significant (chi-square test; p = 0.555).
In the larger case series, Hanna et al. and Tang et al. reported 20% revision rates (n = 15, follow-up 53 months and 55 months, respectively) for custom prosthesis, Athwal et al. reported no revisions for TEA (n = 20, follow-up 34 months), and Capanna et al. reported five-year implant survival of 94% (n = 27) for EPR.14,17,18
Re-operation for local recurrence
The local recurrence rate for all patients was 13% (n = 17). The recurrence rate was highest for TEA (21%), followed by custom prosthesis (13%) and EPR (7%). The incidence of reoperation amongst those cases of local recurrence was 65% (n = 11) with six cases (36%) requiring an above elbow amputation. The alternative treatment options included proximal humeral allografts (12%), soft tissue excisions when recurrence was in the soft tissues (12%), prosthetic replacement (6%) or non-operative palliative management (35%).
Study quality
All studies in this systematic review were non-comparative case series with evidence level 4. The studies were assessed for level of quality using the MINORS criteria. The mean MINORS score for all 11 studies was 9.8 (range 7–11) suggesting that these studies were of poor methodological quality.
Discussion
The most important finding of this study is that although the overall revision rate was 14% at a mean of 44 months, the revision rate for implant failure after TEA appeared to be lower (3%) than after modular EPR (20%) and custom implants (18%) despite similar follow-up periods.
TEA is indicated for alternative pathologies like rheumatoid arthritis, osteoarthritis, acute trauma and post-traumatic arthritis, and revision rates of 15% have been reported in the literature at a 7.8-year follow-up. 30 The highest revision rates were reported after trauma sequelae (30%), while the more common indications like inflammatory arthropathy (11%–13%) and acute trauma (10%–11%) were lower. 31 The literature is generally scarce for EPR and custom prosthesis undertaken for non-oncological aetiology of the elbow joint, as a primary procedure. Hence, it is difficult to make direct comparisons of revision rates after EPR and custom implants for tumours against non-tumour indications.
Cross et al. published their results of 14 custom-made elbow replacements (10 Juvenile Rheumatoid Arthritis[JRA] and 4 Rheumatoid Arthritis [RA]) with mean age 28 years and mean follow-up of 18 years. 32 They noted no revisions but recorded bushing changes and implant removal for infection. Ross et al. performed nine EPRs for fractures with a mean follow-up of six years. 20 Although no revisions were performed, implant removal was recorded for infection (33%). Funovics et al. performed EPR in 38 tumour patients and 15 non-tumour patients. 24 The tumour group saw a 5% revision rate, while the non-tumour group experienced a 20% revision rate. They concluded that EPR was a viable option in patients with a tumour but highlighted the need for careful patient selection in non-tumour patients. It is noteworthy that the non-tumour group included five patients with failed primary TEA.
The familiarity of using a standard TEA implant may be one potential reason for a reduced revision rate with this implant, and although the UK National Joint Registry only reports an average of 467 cases performed annually over the last five years, it is likely that the BESS-GIRFT initiative will improve the experience of surgeons.8,33 In addition, tumour cases which are deemed amenable to TEA rather than EPR or custom implants may require less soft tissue resection and present less challenging surgical procedures.
The local recurrence rate is reported to be higher after TEA (21%) when compared to modular EPR (7%) and custom implant (13%) in this review. This raises concerns that in an effort to use a standard TEA implant the margins of resection maybe compromised, increasing risk of local recurrence. However, the heterogeneity of patients included within these studies and the lack of comparative studies means this is only a hypothesis based on the current evidence.
The overall complication rate for all types of implants in this review was 28% (TEA 33%, modular EPR 29% and custom 25%), with the most common complications being aseptic loosening (11%) and nerve injury (10%). Humeral loosening was three times more common than ulna loosening. 14% of nerve injuries were permanent. The literature suggests that these complication rates are comparable to those reported for alternative indications. The complication rates after TEA for inflammatory arthropathy is reported at 5.2%–30.9%, and after acute trauma or trauma sequelae is at 14.2%–50%. 34 The commonest complications after TEA in inflammatory arthropathy are bushing wear (23%) and deep infection (8%), 33 while in acute trauma they are nerve injury (8%) and aseptic loosening (2.3%), and for trauma sequelae, they are aseptic loosening (18.9%) and infection (5.7%). 35 Cross et al. reported complication rates of 36% for custom elbow prosthesis, with the commonest being bushing wear (28.6%) and deep infection (7%). 32 Ross et al. reported complication rates of 56% for their series of EPRs, with the commonest being deep infection (33.3%) and aseptic loosening (22.2%). 20 Funovics et al. reported similarly high complication rates of 60% for their non-tumour patient group who received EPRs compared to their tumour group who experienced 24% complication rates. 24
This review of tumour patients showed an overall post-operative arc of motion of 90° in the TEA group, 105° in the EPR group and 95° in the custom group. This is compared to the literature which reports 115° for rheumatoid arthritis, 97° for acute trauma and 94° for trauma sequelae when considering TEA prosthesis.35,36,37 With regards to non-oncological indications, the mean arc of motion for custom prosthesis was reported at 111° and EPR was 81°.20,32 Funovics et al.’s comparative study showed no significant difference in arc of motion between their tumour group at 97° and their non-tumour group at 96°. 24
Similarly, when considering the post-operative functional scores in this review, the authors report an MEPS of 75, with 56% of patients reporting good or excellent outcomes after TEA for tumours. The literature reports similar functional outcome scores for TEA with MEPS of 81–94 for rheumatoid arthritis, MEPS of 83–99 for acute trauma and MEPS of 80–94 for trauma sequelae. 34 Tumour patients receiving EPR in this review reported MEPS of 84, MSTS of 80 and TESS of 69. The Inglis–Pellicci score reported by Funovics et al. on EPR for their tumour group was 84, better than the score for their non-tumour group at 76. 24 The authors were unable to find any other scores for EPR in non-tumour patients in the literature, to compare with the tumour cohort in this review. The MEPS of 91 for custom prosthesis in non-tumour patients was better than the custom prosthesis MEPS of 84 in this review. Other functional scores recorded for custom prostheses from this review were MSTS of 79 and TESS of 72, which are comparable to the scores for the EPR cohort in this review.
Limitations of this systematic review are acknowledged and highlighted by the MINORS scores of studies ranging from 7 to 11. The included studies provide only level IV evidence with common weaknesses being the low study numbers, retrospective nature of the studies and the lack of comparative groups. Heterogeneity of the underlying tumour diagnosis and reporting of outcome measures precludes data synthesis and restricts direct comparison of results. Determining post-operative improvements proved difficult as this requires pre-operative measurements which were commonly unavailable. The studies included were published from 1999 to 2019; no time filter was applied to the search in order to improve the volume of studies and patients available for analyses. However, this meant that the effect of evolving and improving surgical technique is difficult to factor into the overall outcomes. The range of patient follow-up is also very large (1–372 months) and the true long-term outcomes of elbow arthroplasty for tumours are therefore hard to quantify.
This systematic review has identified a significant demand for high-quality prospective studies in order to better report and compare the outcomes of different elbow arthroplasty options in oncological patients.
Conclusions
Elbow prosthesis reconstruction after tumour resection can provide good functional outcomes at mid-term follow-up. However, there is a high overall complication rate of 28% and a revision rate of 14%. Although high, the complication and revision rates are comparable to other indications for elbow arthroplasty including rheumatoid arthritis (which is the commonest indication), acute trauma and traumatic sequelae. However, in order to effectively, scientifically compare the functional outcome of different elbow arthroplasty options amongst both the tumour and non-tumour groups, we conclude that more robust prospective studies are required.
It is recommended that all patients are discussed at a Multi-Disciplinary Meeting that includes a specialist elbow reconstructive surgeon as well as a surgeon with an oncological interest. There should be robust surveillance methods and audits with data collection that includes outcome scores.
Footnotes
Acknowledgements
The authors would like to thank the Frances-Costello library for their assistance with the electronic literature search.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, authorship, and/or publication of this article.
Guarantor
SMH.
Contributorship
GM –PRISMA process, data extraction and manuscript first draft; RWJ – manuscript first draft; GO – PRISMA process; MSC – data extraction from articles; PC – statistics, revision manuscript and SMH – conception and manuscript final review
