The Functional Outcomes and Complications of Different Reconstruction Methods for Giant Cell Tumor of the Distal Radius: Comparison of Osteoarticular Allograft and Three-Dimensional-Printed Prosthesis

doi:10.21203/rs.2.17169/v1

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The Functional Outcomes and Complications of Different Reconstruction Methods for Giant Cell Tumor of the Distal Radius: Comparison of Osteoarticular Allograft and Three-Dimensional-Printed Prosthesis

https://doi.org/10.21203/rs.2.17169/v1

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published 03 Feb, 2020

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Background: En bloc excision is increasingly used for the management of giant cell tumor in the distal radius. Osteoarticular allograft is extensively used for decades, also custom-made prosthesis reconstruction is recently applied widely. We aimed to firstly compare the clinical outcomes of the two procedures.

Methods: We retrospectively analyzed thirty-two patients with Campanacci III or recurrent GCT of the distal radius for follow-up at a mean of 33.2 months. Sixteen underwent osteoarticular allograft reconstruction (allograft group) and sixteen received cementless three-dimensional(3D)-printed prosthesis reconstruction (prosthesis group) between March 18, 2013, and May 20, 2018. All patients were assessed by clinical and radiological examinations, including pre- and postoperative active range of motion (ROM) of the wrist, VAS score, grip strength, degenerative change of wrist, Mayo wrist score and Musculoskeletal Tumor Society (MSTS) score. Complications were evaluated on the Henderson classification.

Results: Both groups showed significantly increased ROM, grip strength, Mayo score and MSTS score postoperatively. MSTS and Mayo score were significantly higher in prosthesis group. There was no significant difference in grip strength and VAS between two groups. In allograft group, one had a local recurrence; one had a late infection; four had wrist subluxation. All patients had degenerative changes (mean 9 months, 95% CI: 8.03-9.97). One had resorption of allograft without allograft bone fracture. In the prosthesis group, one had a local recurrence. Three developed wrist subluxation. Three had separation of the distal radioulnar joint. None of the patients developed degeneration of wrist.

Conclusions: Our study firstly compared the objective functional outcomes and complications of two reconstructive methods for the Campanacci III or recurrent GCT in the distal radius. 3D-printed prosthesis replacement can preserve better wrist function than allograft reconstruction at short-term. During 3D-printed prosthesis design, preoperative morphological assessment of affected proximal row carpal is helpful to control the postoperative dislocation. After allograft reconstruction, degeneration of wrist, which has been proved in all patients, severely influence their wrist function. Therefore, compared with allograft reconstruction, 3D-printed prosthesis reconstruction shows its irreplaceable advantages at early-stage application, especially in wrist function, but its larger cases research at longer follow-up are needed.

Orthopedic Surgery

distal radius

giant cell tumor

osteoarticular allograft

3D-printed prosthesis

The distal radius is the third most common location for giant cell tumor (GCT) after the distal femur and proximal tibia, which 10% of GCT involve the distal radius[1, 2]. Although the majority do not support this theory that GCT in the distal radius is more aggressive, controversy exists on surgical options in patients with GCTB in the distal radius and its recurrence. Intralesional curettage and cement packing is the most common treatment for Campanacci I and II lesions[3, 4]. However, En bloc resection of Campanacci III or recurrent GCT of the distal radius with reconstruction is associated with a lower risk of local recurrence and similar functional outcomes than intralesional surgery[3, 5–7]. However, the distal radius is a fairly special anatomic site for GCT, with limited soft tissue coverage and bone mass, which has shown one of the highest recurrent rates after intralesional surgery[4, 6, 8]. For most patients with Campanacci Grade III or recurrent GCT, the local recurrent rate is most concerned. Also, repeated recurrence means increasing the operating difficulty and reducing the possibility of limb salvaging.

Reconstruction of the wrist joint following en bloc resection of the distal radius is challenging because of the high functional demands of the wrist, the limited surrounding soft tissue, and the proximity of important nerves and tendons[9]. Numerous reconstructive procedures have been described including prosthetic replacement[9–11], osteoarticular allograft[12, 13], allograft fusion[14], arthrodesis using bulk autograft[1, 2], ulnar translocation[15], non-vascularised[16] or vascularised[17] fibular graft with or without arthrodesis[18]. Although these techniques all come with their unique advantages and evitable complications, a gold standard for distal wrist reconstruction has not been established.

As a major treatment method, osteoarticular allograft has its own advantages, including biologic scaffold, osteoinduction and relatively appropriate anatomical match for host proximal row carpel[13, 19]. However, the use of osteoarticular allograft is extremely restricted by the limited quantity and severe complications, including ankylosis, rejection, and allograft fracture[19]. Therefore, several prosthetic arthroplasties were reported in the last decade, and the acceptable results of prosthesis reconstruction were demonstrated in short- to mid-term[9, 20, 21]. However, their potential complications included aseptic loosening caused by cement-fixation and inappropriate anatomical match[11, 22]. Following the technological advances in materialogy and manufacturing, 3-dimensional(3D) printing technology has been introduced to the region of orthopedics. Our institution reported the first study of the custom-made cementless 3D-printed prosthesis for distal radius GCT[21].

Despite experience with osteoarticular allograft and prosthetic replacement GCT of the distal radius, there is no report regarding the comparative study of functional results and complications have been published ever before. The purpose of this study was to investigate mid-term clinical outcomes of patients with GCT in the distal radius after these two different reconstructions.

Patients

The study was conducted retrospectively on patients treated between March 18, 2013, and May 20, 2018, in our institution. The inclusion criteria included: 1) histologically proven GCT with needle biopsy; 2) Campanacci III or recurrent giant cell tumor of the distal radius with the cortex or soft tissue invasion; 3) since 2013, patients, who rejected arthrodesis using allograft and/or ulnar translocation, underwent allograft reconstruction or prosthesis reconstruction. Exclusion criteria consisted of GCT in the distal radius with metastasis and any other surgical procedure. According to the reconstructive methods, we considered consecutive series in our institution with GCT of the distal radius treated with osteoarticular allograft (allograft group) and 3D-printed prosthesis (prosthesis group).

The preoperative assessments included: radiographs of bilateral forearm and wrist, computed tomography (CT) scan of the chest, bilateral forearm and wrist, magnetic resonance imaging (MRI) of the affected side and optional total body bone scan. All patients were evaluated for pain according to a 10-cm VAS score, the range of motion (ROM) recorded by using a goniometer, grip strength of bilateral wrist joint, Mayo wrist score[23] and Musculoskeletal Tumor Rating Scale (MSTS)[24] of the wrist, preoperatively and postoperatively. All patients were assessed every 3 months during the first year of follow-up and every 6 months thereafter with a physical examination, VAS, functional evaluation of the wrist, radiographs of the wrist and chest and the degenerative changes of the wrist[25]. This study was performed according to the principles embodied in the Declaration of Helsinki and the Institutional Review Board of Sichuan University West China Hospital. Written informed consent was obtained from all patients when they began treatment for osteoarticular allograft or 3D-printed prosthesis. The study protocol followed all appropriate guidelines according to the Declaration of Helsinki.

Allograft preparation and Prosthesis design

In the allograft group, to obtain the reconstruction as anatomical as possible, the isometric x-rays of the bilateral side and 3D-CT scan were taken. Fresh-frozen allografts were supplied by the bone-bank facility without soft tissue (West China Hospital, Chengdu, Sichuan).

In the prosthesis group, all prostheses were custom-made designed for each patient by our team and produced (Chunli, Beijing, People’s Republic of China). Based on our previous study[21] and experience, the design has been modified and improved for years with the help of Mimics V17.0 software (Materialise Corp., Leuven, Belgium). The main components of the 3D-printed prosthesis were mainly composed of an ultrahigh-molecular-weight polyethylene (Orthoplastics Ltd., Lancashire, UK), repairing pores for soft tissue reconstruction, shaft and stem coated with hydroxyapatite (titanium alloy)(Figure 1). All prostheses were printed by electron beam melting technology (ARCAM Q10, Mölndal, Sweden). It took 2 or 4 weeks to manufacture the prosthesis, during which time the patients were treated with NSAIDs if necessary.

Surgical technique and Postoperative management

Patients were submitted to general anesthesia. The tumor was resected en bloc through a dorsal approach including the previous biopsy and operating incision. Soft tissue dissection, including wrist ligaments, capsule, triangular fibrocartilage complex (TFCC), biopsy track and hematoma, was dependent on the tumor border. Step-cut osteotomy was performed with a safe margin (1.5cm away from the tumor border) according to X-rays and MRI. In allograft group, an appropriate LCP was fixed to the allograft bone and the remaining radius to obtain initial stability and avoid rotation of the distal radius Allograft bone was fixed with a suitable locking compression plate (LCP, Wego, Shandong, People’s Republic of China) to bridge the allograft and the remaining radius[13]. In prosthesis group, stable fixation between the prosthesis and reamed radius canal was enhanced by press-fitting the distal stem. The remaining dorsal and/or palmar ligaments, joint capsule and TFCC were sutured to the allograft bone or repairing pores of the prosthesis.

Based on the reserved soft tissue, an above-elbow cast was administered to patients with massive resection. After a four-week immobilization, active wrist exercises were acceptable.

Statistical analysis

Survival data were compiled using Kaplan-Meier analysis. Prosthesis survivorship was determined for implants using revision or removal of the components for any reason as an end point, according to Henderson et al[26]. No complication survivorship was defined for the patient without wrist pain, degeneration of the wrist, subluxation, and separation of the distal radioulnar joint at the last follow-up.

The normality of the continuous data was verified by the Shapiro-Wilk test. Normally distributed parameters and abnormally distributed parameters were assessed by the independent sample t-test and the Mann–Whitney U test, respectively. A p-value of 0.05 or less was examined as statistically significant. Comparisons were conducted between allograft and prosthesis group by log-rank test.

Patient Characteristics

From March 18, 2013, and May 20, 2018, 32 consecutive patients were enrolled. All pathology materials were confirmed at West China Hospital. The clinical characteristics of the two groups and their comparison are presented in Table 1. When compared, patients of the two treatment groups had a significant difference in gender, age, length of resection and follow-up time.

Functional Outcomes

There was no significant difference between the two groups preoperatively in the functional outcomes. In the allograft group, there was a significant increase in ROM except for extension postoperatively. The mean ROM of the wrist postoperatively, was 37 active extension (range, 20-65), 27 flexion (range, 10-50), 51 pronation (range, 30-65), and 55 supination (range, 40-80). MSTS score of wrist and Mayo wrist score was significantly increased postoperatively, averaged 71.6% (57.1-82.9%) and 66.6 (60-75) respectively. Postoperatively grip strength was 54.8% (range, 44.4-70.8%) of the normal contralateral hand compared with 28.9% (range, 13.8-38.3%) preoperatively, with a significant increase. Relates to the VAS score, it was 5.6 (range, 4-7) preoperatively and 1.2 (range, 0-3) postoperatively (Table 2).

In the prosthesis group, with regards to the ROM, there were significant differences between pre- and post-operatively. It was 61 active extension (range, 20-85), 44 flexion (range, 20-80), 54 pronation (range, 30-80), and 59 supination (range, 25-85) after prosthesis reconstruction (Figure 2). With respect to the MSTS score of wrist and Mayo wrist score, there was a significant increase after the operation, respectively averaged 81.7% (60-88.6 %) and 71.3 (35-85). There were significant differences between pre- and post-operatively in grip strength. It was 33.4% (range, 11.1-54.5%) and 61.5% (range, 27.7-76%) separately. And the VAS score was 5.6 (range, 4-7) preoperatively and 1.3 (range, 0-4) postoperatively. This difference was statistically significant. (Table 3)

When compared to the variations of functional outcomes before and after surgery, although prosthesis group showed a better of ROM in extension, flexion pronation and supination, only extension (7.2 vs 29.7, p=0.01) and flexion (1.3 vs 12.5, p=0.020) were statistically significant. There was a significant difference in the variation of Mayo wrist score (48.8 vs 51.6, p=0.047) in two groups. The variation of MSTS score was no difference between the osteoarticular allograft and the prosthesis groups (23.4% vs 27%, p=0.342), as well as grip strength (25.9% vs 28.2%, p =0.56) and VAS score (4.4 vs 4.3, p =0.89) (Table 4).

When compared to the variations of satisfaction before and after surgery, there was no significant difference in the variation of satisfaction in Mayo wrist score (20.9 vs 21.6, p=0.726) in two groups. And there was a significant increase in the variation of emotional acceptance in MSTS score (4.1 vs 1.4, p<0.01) in the prosthesis group. There was a significantly lower pain in prosthesis group with both two Mayo (25.00 vs 21.88, p<0.01) and MSTS (5.00 vs 4.38, p=0.02) score. Although the MSTS score of 2-year follow-up was higher than the latest follow-up, there was no significant difference (57.68 vs 57.33, p=0.872). There was no significant difference between MSTS score of 2-year follow-up allograft and latest follow-up prosthesis groups.

Complications

In the allograft group, all patients were alive. Of 16 patients, only one had a local recurrence (type 5A) occurred 6 months after surgery, leading in single tumor resection. And one had a late infection (50 months after surgery) caused by plate exposed (type 1B), resulting in debridement and removal of the plate. Four had wrist subluxation (type 1A), respectively occurred 2, 3, 6 and 36 months after surgery. At the lastest follow-up, all patients had degenerative changes (three had Grade 2, fourteen had Grade 3, mean 9 months, range 3-12 months), according to the Knirk and Jupiter scale[25]. One had resorption of allograft (type 2B) without allograft bone fracture. Neither patient had a structural failure, soft-tissue failure, nonunion, metastasis, and pain.

In the prosthesis group, none of the patients died. One of the sixteen patients had a local recurrence (type 5A) occurred 7 months after surgery. Another operation was performed to resect the recurrent tumor, then the patient developed lung metastasis 8 months later without additional therapy. Three patients developed wrist subluxation (type 1A) occurred within one month after surgery. Three had separation of the distal radioulnar joint. Two of the three patients occurred in 1 month after surgery and the other one developed in 7 months after surgery. Neither patient had a structural failure, soft-tissue failure, aseptic loosening, infection, pain, and degenerative changes because of surgery. There was no significant difference between allograft and prosthesis group in complications according to Henderson classification

There was no significant difference between allograft and prosthesis group in implant survival (Figure 3), and the median survival time was not reached in both two groups. Because of no patient was died in the last follow-up, the 3-year OS rate was 100.0% in the allograft group, and the 1-year OS rate was 100.0% in prosthesis group. Median OS has not been reached in both groups (figure 9).

Campanacci III or/and recurrent GCT in the distal radius is characterized by strong invasion and high recurrent rate. The primary goal is an oncologic cure. Further functional satisfaction is intensely demanded. The present study is the first comparing these two techniques for the treatment of GCT in the distal radius. To highlight differences between the 3D-printed prosthesis and allograft reconstruction, the comparison is carried out in functional outcomes and complications. With respect to functional outcome, MSTS and Mayo score was evaluated. In general, the prosthetic group had a significantly higher scores, when compared to the variation of before and after surgery (18.2% vs 27.5%, p=0.017 and 48.8 vs 51.6, p=0.047). Also, prosthetic group had a significant higher score, compared to the postoperative evaluation (Table 4).

Comparison of ROM

For the variation of ROM in Mayo score, there was a significantly higher score in prosthesis group compared with allograft group (3.75 vs 6.56, p=0.043). Nevertheless, there was no significant difference in both two groups with MSTS score (0.63 vs 0.75, p=0.752). This discrepancy in ROM between Mayo and MSTS may be partially explained by that ROM is given a weighting of 25 percent in Mayo score, compared with 14 percent in MSTS system. In addition, most patients get full marks in MSTS system, because the ROM is more than 120, including extension, flexion, supination, pronation and radial and ulnar deviation. But the score of ROM is valued by a percentage of the contralateral side in the Mayo system, which hardly gets full marks.

In previous studies, custom-made cemented prosthesis reconstruction had obtained reasonable ROM, with different types, including distal radial prosthesis [9, 11, 20], total wrist joint prosthesis prothesis[22, 27, 28]. For our 3D-printed uncemented prosthesis, not only the individual and precise design, but also “press-fit” fixation make surgery easy and result in considerable functional outcomes[21]. Our 3D-printed prosthesis has three main advantages. First, a thick and suitable polyethylene liner is made according to the contralateral side; second, the reserved bone crest of the distal radius on the shaft insure appropriate implantation without any rotation; third, seven or eight pores, on distal prosthetic edge, provide sufficient place for soft tissue reconstruction. For allograft group, all patients developed grade 2 or grade 3 degeneration of the wrist joint (Figure 4). And the median degeneration-time was 9 months (95% CI: 8.03-9.97) (Figure 5). There might be one explanation that creeping substitution, the process through which the allograft is gradually replaced by living bone[29], goes to tide mark under dead articular cartilage, therefore there is a risk of subchondral collapse[30]. When respect to forearm rotation, the distal radioulnar joint (DRUJ) plays a critical role. We reveal that four patients developed separation of the DRUJ (Figure 6) in prosthetic reconstruction. Based on the tumor border, most structures of triangular fibrocartilage complex (TFCC) were not preserved in the four patients, therefore there was the tendency for separation of the DRUJ. With respect to the stabilizing structures of DRUJ, it includes the TFCC, the surrounding ligament, tendon, muscle, the interosseous membrane, the bone itself, and the capsule[31]. The TFCC, containing superficial and deep fibers, is the main stabilizer of the DRUJ[32]. Many studies concluded that in pronation the dorsal superficial fibers tighten, as do the deep palmar fibers and vice versa[31]. Therefore, we speculate the relative decrease in pronation and supination is associated with insufficient reconstruction of TFCC. When achieving soft tissue reconstruction, we suggest the retained fibers of TFCC should be precisely reconstructed by suturing. No separation of the DRUJ was detected in allograft patients, because of selection bias and longer immobilization postoperatively.

Comparison of Pain

Compared to the pain postoperatively, there was a significantly lower pain in prosthesis group with both Mayo (25.00 vs 21.88, p<0.01) and MSTS (5.00 vs 4.38, p=0.02) score. However, the postoperative pain score was no difference with VAS evaluation (1.2 vs 1.3, p =0.985). Although the pain was fairly decreased postoperatively in both two reconstruction methods, patients in prosthesis group less complained of pain, compared with allograft group. We speculate that the anatomical and precise design increases matching of joint compatibility and improved comfort for patients. For prosthesis reconstruction, Zhang et al. reported only one patient complained of moderate pain in 11 patients[9], Wang et al. reported no patient suffered pain with activity in 10 patients[20]. Secondly, allograft reconstruction has shown a high rate of joint degeneration, which increased the level of pain and decreased the ROM. Rabitsch et al. reported 100% joint degeneration in 4 patients[33], Duan et al. reported 100% joint degeneration in 15 patients[13].

Comparison of Satisfaction

With respect to satisfaction postoperatively with MSTS score, there was a significantly higher satisfaction in prosthesis group compared with allograft group (2.88 vs 4.25, p<0.01). Nevertheless, there was no significant difference in both two groups 22.19 vs 23.44, p=0.30) with Mayo score. This discrepancy may be partially explained by that the difference in weighting of satisfaction in Mayo and MSTS score. Although the discrepancy was found in Mayo and MSTS evaluating functional outcomes, a comprehensive comparison was performed by Mayo and MSTS score between two groups. Overall, the prosthetic reconstruction had a better functional outcome, compared with allograft reconstruction.

Comparison of Complications With Henderson Classification

With respect to complications, the main potential problems for allograft, including nonunion, allograft fracture, wrist osteoarthritis, slow incorporation of the allograft, and rejection, have been reported after en bloc resection (Table 5). Bus et al’s compared complication rates of allograft reconstruction between different sites, the distal radius showed a significantly lower risk in structural failure and infection, compared with the proximal tibia, distal femur, and proximal humerus[38]. Also, LCP makes reconstructions easy and may be expected to fewer complications[13]. Therefore, previous authors have suggested that if an intercalary allograft survives the critical three to four years, it is likely to last for many years[39]. In our study, four had palmer subluxation. Three of the four patients developed palmer subluxation within 6 months after surgery. There might be three reasons: firstly, the strength of flexor is greater than extensor[31], developing the tendency of palmer dislocation; secondly, all the patients underwent a dorsal approach, protecting most of stabilizing structures in palmar; thirdly, without the pores in the prosthesis, the retained soft tissue suturing is tedious and unreliable. One patient progressively became palmer subluxation in the third year after operation (Figure 7). According to the radiograph, we speculate subluxation was subordinate to the carpal degeneration.

The main potential complications associated with the prosthesis are subluxation, aseptic loosening, infection, soft-tissue failure and fracture of the prosthesis[9, 11, 20, 22, 40] (Table 6). In the present study, one had a soft tissue recurrence and later developed lung metastasis. Three patients developed palmar subluxation occurred within one month after surgery (Figure 8). Revision surgery is inevitable for the patient. In theory, mirroring of the contralateral normal distal radius is the best anatomical match. Accounting distal radial volar palmar tilt 11-12[31], the tendency, for volar subluxation for the carpus to slide off the distal radius, is inevitable. However, soft-tissue tension is affected by the expansive growth of the lesion, especially in Campanacci III or recurrent GCT of bone[42]. And the degeneration of proximal row carpal is generally detected in our elder or recurrent patients. Hence, totally mirroring of the contralateral side may mislead us. We propose that the degeneration of proximal row carpal should be religiously considered. Additionally, retained soft tissue was advised to sequentially reconstruct for appropriate soft tissue tension[21]. What’s more, radius lengthening combined with folding-plasty of soft-tissue reconstruction is an effective method.

Most study report wrist joint degeneration after allograft reconstruction in table 5, while wrist joint degeneration was rarely detected in prosthesis method. Duan et al. reported degeneration was revealed in a mean of 4 months postoperatively[13]. We found degeneration with a mean of 9 months. We speculate that the wrist joint degeneration secondary to allograft reconstruction develops in the first year postoperatively. With respect to comparison of wrist subluxation, although there was no significant difference between allograft (4 patients) and prosthesis (3 patients) groups, different mechanisms were found in two groups. For allograft reconstruction, subluxation was subordinate to the gradual carpal degeneration. While subluxation was mostly depended on prosthetic design and retained soft tissue.

This study presents several limitations, mainly due to the nature of the disease. First of all, our study was a retrospective with a small sample (16 and 16 patients in the allograft and prosthesis group, respectively). As such, our small sample size may be expected to result in low statistical power. Second, the follow-up time was significantly different in the two groups, also the follow-up is insufficient to make long-term results. Thirdly, no patient was administered denosumab preoperatively. The efficacy of denosumab has been demonstrated in patients with unresectable or recurrent GCT of bone, according to NCCN guideline and previous studies[43]. However, denosumab did not show any effect on reducing recurrent rate[44], also complications such as sarcomatous transformation should be aware[45]. Fourthly, our findings are only based on the respective data in our institution. This implies a study selection bias that must be acknowledged. Finally, we also did not have sufficient reconstruction types such as autograft fibula grafts used either for arthrodesis or osteoarticular reconstructions nor did we look at vascularized fibular grafts, so our ability to state that arthrodesis is a superior reconstruction method is limited. We can show only that the results in our patients provided them with reasonable function.

This is the first study comparing the objective functional outcomes and complications of two reconstructive methods for the Campanacci III or recurrent GCT in the distal radius. Despite including subluxation cases, 3D-printed prosthesis replacement at short-term follow-up still can preserve better wrist function than osteoarticular allograft reconstruction at short-term even median-term follow-up. During 3D-printed prosthesis design, preoperative morphological assessment of affected proximal row carpal is helpful to control the postoperative dislocation. In addition, considering relatively instability of joint capsule reconstruction, properly delayed rehabilitation is recommended. After osteoarticular allograft reconstruction, degeneration of wrist, which has been proved in all patients, severely influence their wrist function rather than that of the patients with prosthesis reconstruction. Therefore, compared with osteoarticular allograft reconstruction, 3D-printed prosthesis reconstruction shows its irreplaceable advantages at early-stage application, especially in reconstruction of wrist function, but its larger cases research at longer follow-up are still needed.

GCT: giant cell tumor; 3D: three-dimensional; DRUJ: distal radioulnar joint; ROM: range of motion; TFCC: triangular fibrocartilage complex; MSTS: musculoskeletal tumor society.

Acknowledgements

We are thankful for the support of the nurse team from the Department of Orthopedics, West China Hospital. We are thankful for the patients enrolled in this study.

Funding

This work was supported, in part, by the National Natural Science Foundation of China (81702664) and Chengdu science and technology project (2017-CY02-00032-GX).

Availability of data and materials

The datasets used and analyzed during the current study are available from the corresponding author on reasonable request.

Authors’ contributions

TYW, LM, and CQT were involved with the concept and design of this manuscript. YZ, YL and FT were involved with the acquisition of subjects and data. MXL, JW, YQZ, and CQT were involved in the preoperative 3D design. YTW, LM and CQT were involved in postsurgical evaluation of the patients. All authors contributed toward data analysis, drafting and critically

revising the paper, gave final approval of the version to be published, and agree to be accountable for all aspects of the work.

Authors’ information

Department of Orthopedics, West China Hospital, Sichuan University, Guoxue Xiang #37, Chengdu, Sichuan 610041, People’s Republic of China.

Ethics approval and consent to participate

This study was approved and monitored by the Ethical Committee of West China Hospital, Sichuan University in China (No.2019117). All patients signed the informed consent.

Consent for publication

Written informed consent was obtained from all patients for publication of this study and any accompanying images.

Competing interests

The authors report no competing interest in this work.

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28.Damert HG, Altmann S, Kraus A: Custom-made wrist prosthesis in a patient with giant cell tumor of the distal radius. Arch Orthop Trauma Surg 2013, 133(5):713–719.

29.Aho AJ, Ekfors T, Dean PB, Aro HT, Ahonen A, Nikkanen V: Incorporation and clinical results of large allografts of the extremities and pelvis. Clinical orthopaedics and related research 1994(307):200–213.

30.Campanacci L, Manfrini M, Colangeli M, Ali N, Mercuri M: Long-term results in children with massive bone osteoarticular allografts of the knee for high-grade osteosarcoma. J Pediatr Orthop 2010, 30(8):919–927.

31.Haugstvedt JR, Langer MF, Berger RA: Distal radioulnar joint: functional anatomy, including pathomechanics. The Journal of hand surgery, European volume 2017, 42(4):338–345.

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33.Rabitsch K, Maurer-Ertl W, Pirker-Fruhauf U, Lovse T, Windhager R, Leithner A: Reconstruction of the Distal Radius following Tumour Resection Using an Osteoarticular Allograft. Sarcoma 2013, 2013:318767.

34.Smith RJ, Mankin HJ: Allograft replacement of distal radius for giant cell tumor. The Journal of hand surgery 1977, 2(4):299–308.

35.Cheng CY, Shih HN, Hsu KY, Hsu RW: Treatment of giant cell tumor of the distal radius. Clinical orthopaedics and related research 2001(383):221–228.

36.Asavamongkolkul A, Waikakul S, Phimolsarnti R, Kiatisevi P: Functional outcome following excision of a tumour and reconstruction of the distal radius. Int Orthop 2009, 33(1):203–209.

37.Li J, Jiao Y, Guo Z, Ji C, Wang Z: Comparison of osteoarticular allograft reconstruction with and without the Sauve-Kapandji procedure following tumour resection in distal radius. J Plast Reconstr Aesthet Surg 2015, 68(7):995–1002.

38.Bus MP, van de Sande MA, Taminiau AH, Dijkstra PD: Is there still a role for osteoarticular allograft reconstruction in musculoskeletal tumour surgery? a long-term follow-up study of 38 patients and systematic review of the literature. Bone Joint J 2017, 99-B(4):522–530.

39.Ortiz-Cruz E, Gebhardt MC, Jennings LC, Springfield DS, Mankin HJ: The results of transplantation of intercalary allografts after resection of tumors. A long-term follow-up study. J Bone Joint Surg Am 1997, 79(1):97–106.

40.Gold AM: Use of a Prosthesis for the Distal Portion of the Radius Following Resection of a Recurrent Giant-Cell Tumor. J Bone Joint Surg Am 1965, 47:216–218.

41.Gokaraju K, Sri-Ram K, Donaldson J, Parratt MT, Blunn GW, Cannon SR, Briggs TW: Use of a distal radius endoprosthesis following resection of a bone tumour: a case report. Sarcoma 2009, 2009:938295.

42.Campanacci M, Baldini N, Boriani S, Sudanese A: Giant-cell tumor of bone. J Bone Joint Surg Am 1987, 69(1):106–114.

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45.Tsukamoto S, Righi A, Vanel D, Honoki K, Donati DM, Errani C: Development of high-grade osteosarcoma in a patient with recurrent giant cell tumor of the ischium while receiving treatment with denosumab. Japanese journal of clinical oncology 2017, 47(11):1090–1096.

Table 1 Demographic and radiographic characteristics of the two treatment groups.

Characteristic	Osteoarticular allograft group			3D-printed prosthesis group			p-value
	Mean	SD	Range	Mean	SD	Range
Number of patients	16	-	-	16	-	-	-
Male/female	9/7	-	-	6/10	-	-	0.48
Age (years)	36.7	12.0	24-63	39.7	11.8	21-65	0.32
Side (right/left)	7/9	-	-	9/7	-	-	0.72
Primary/Recurrence	12/4	-	-	11/5	-	-	>0.999
Length of resection (cm)	8.1	1.0	7-10	5.3	1.7	3.5-9	<0.001
Follow-up (months)	35.0	17.1	10.7-64.4	31.4	11.4	13.7-48.2	0.242

Table 2 preoperative/postoperative details in allograft group

Patient

number

Age (years)/

gender

Campanacci

grade

Follow-up

(months)

Range of motion

Grip strength(% of normal side)(%)

VAS

score

Degenerative grade/ Occurrence time after operation(month)

Mayo score

MSTS

Extension (°)

Flexion (°)

Supination (°)

Pronation (°)

total

motion

Extension (°)

Flexion (°)

Supination (°)

Pronation (°)

Functional

Activity

Emotional

Acceptance

40/M

III

64.4

30/30

20/25

30/55

40/40

28.9/54.8

6/0

III/9

20/65

48.6/77.1

5/5

1/5

5/5

1/3

1/5

38/M

Recurrent

61.8

40/65

25/30

35/55

40/55

22.0/70.8

5/2

III/12

25/65

60.0/65.7

5/5

1/5

5/5

3/5

1/3

5/3

27/M

III

60.3

35/20

30/30

45/60

35/30

18.6/60.0

6/1

III/9

15/65

42.9/77.1

5/5

1/5

5/5

3/5

1/5

1/3

33/M

Recurrent

20/25

30/20

25/50

35/40

38.3/50.0

7/1

III/6

15/60

48.6/57.1

3/5

1/5

5/5

5/3

1/3

1/0

1/1

25/M

III

38.2

35/65

30/50

30/80

35/65

13.8/55.0

5/1

III/12

10/75

60.0/82.9

5/5

1/5

5/5

1/3

3/3

1/5

29/M

Recurrent

33.1

35/65

40/25

20/65

25/55

21.4/61.1

6/1

III/12

15/75

48.6/71.4

5/5

1/5

5/5

1/3

3/3

1/3

25/M

III

13.1

20/25

10/20

20/40

15/55

32.9/53.7

6/0

III/3

20/70

60.0/65.7

3/5

1/5

5/5

3/3

3/1

1/1

62/F

III

10.7

35/30

40/35

35/60

40/60

33.2/53.8

5/1

III/12

20/70

54.3/77.1

5/5

1/5

5/5

1/3

37/F

III

27.9

45/25

45/45

45/65

30.0/53.3

6/1

III/6

20/70

54.3/77.1

5/5

1/5

5/5

1/3

3/3

35/M

III

24.5

20/25

15/35

35/50

25/55

33.0/44.4

4/1

III/9

15/60

60.0/65.7

3/5

1/5

5/5

3/3

1/3

27/F

III

22.5

30/30

25/20

40/45

33.0/54.2

5/1

III/12

20/65

60.0/71.4

5/5

1/5

5/5

1/3

3/3

1/3

63/F

III

20.9

20/25

20/10

30/45

33.0/54.5

5/3

III/6

15/60

42.9/71.4

3/5

1/5

5/3

1/3

3/3

1/3

24/F

III

25/30

25/25

35/55

40/35

33.1/52.0

6/2

III/8

20/65

65.7/71.4

5/5

1/5

5/5

3/3

1/3

3/3

35/F

Recurrent

25/40

5/25

20/50

35/55

33.1/55.6

7/2

III/12

15/65

45.7/71.4

3/5

0/5

5/5

3/3

1/3

45/M

III

30/50

25/25

50/65

35/50

25.0/54.8

4/1

II/9

20/70

54.3/77.1

5/5

3/5

5/5

1/3

42/F

III

35/45

20/25

45/65

30/60

33.3/48.4

6/1

II/12

20/65

48.6/65.7

5/5

1/5

5/5

1/3

1/1

Table 3 preoperative/postoperative details in prosthesis group

Patient

number

Age (years)/

gender

Campanacci

grade

Follow-up

(months)

Range of motion

Grip strength(% of normal side)(%)

VAS

score

Degenerative grade/ Occurrence time after operation(month)

Mayo score

MSTS

Extension (°)

Flexion (°)

Supination (°)

Pronation (°)

Total

motion

Pain

Stability

Deformity

Strength

Functional

Activity

Emotional

Acceptance

42/F

III

48.2

25/45

40/40

25/75

40/80

11.1/27.7

6/0

None

30/60

31.4/88.6

3/5

1/5

3/5

0/3

1/5

45/M

Recurrent

45.7

45/85

25/35

50/80

30/50

33.3/53.3

5/2

None

20/80

51.4/77.1

5/5

1/3

3/3

5/5

3/3

1/3

0/5

34/F

III

45.3

40/60

30/40

45/60

30/45

25.0/55.0

6/1

None

20/75

45.7/88.6

5/5

1/5

3/3

5/5

3/3

1/5

0/5

37/M

Recurrent

35.4

10/60

30/45

20/50

30/50

40.0/63.8

7/1

None

15/75

51.4/82.9

3/5

1/5

5/3

5/5

3/3

1/5

0/3

45/F

III

34.7

50/65

70/80

20/50

30/50

37.0/64.3

5/1

None

20/75

57.1/82.9

5/5

1/5

5/3

5/5

3/3

1/3

0/5

46/M

Recurrent

33.8

30/80

40/60

20/50

30/50

42.2/62.5

6/1

None

15/85

57.1/82.9

5/5

1/3

5/3

5/5

3/3

1/5

0/5

65/F

III

31.8

15/45

10/30

15/40

5/40

23.8/59.1

6/0

None

20/70

48.6/65.6

1/5

3/3

5/5

5/3

0/3

3/1

0/3

37/F

III

31.7

50/75

40/50

50/60

45/55

28.0/70.8

5/1

None

20/75

57.1/82.9

5/5

1/5

5/3

3/3

1/5

0/5

27/F

III

30.7

60/75

60/60

50/65

50/70

26.9/66.7

6/1

None

25/75

54.3/88.6

5/5

1/5

5/5

1/3

1/5

24/F

III

26.3

15/50

10/45

30/60

20/60

33.3/66.7

4/1

None

15/70

51.4/82.9

3/5

5/3

5/5

1/3

0/5

21/F

III

17.9

30/85

30/30

45/85

45/55

16.0/66.7

5/1

None

30/85

51.4/88.6

5/5

1/5

5/5

1/3

1/5

0/3

45/M

III

42.4

15/20

20/20

20/25

20/30

36.4/46.3

5/4

None

15/35

62.9/60.0

3/3

5/3

5/1

3/3

0/5

45/F

III

36.7

20/70

30/50

45/60

30/35

45.8/76.0

6/2

None

15/80

57.1/82.9

5/5

1/3

5/5

3/3

1/3

0/5

56/M

Recurrent

14.4

10/55

5/40

20/60

30/60

44.4/66.7

7/2

None

10/70

54.3/88.6

3/5

0/5

5/5

3/3

0/5

25/F

III

14.4

50/65

40/50

50/70

40/60

35.7/66.7

4/1

None

20/75

57.1/82.9

5/5

3/5

5/5

3/3

1/3

0/3

41/M

Recurrent

13.7

30/35

25/30

50/50

40/50

54.5.72.7

6/1

None

25/55

57.1/60.0

5/5

1/5

5/3

3/3

1/1

0/1

Table 4 Statistical comparison of clinical results between the two treatment groups.

Measure	Osteoarticular allograft group (N = 16)	3D-printed prosthesis group (N = 16)	p-value allograft vs prosthesis
Postoperative
Extension (°)	37.2 (range 20~65)	60.6 (range 20~85)	<0.01
Flexion (°)	26.6 (range 10~50)	44.1 (range 20~80)	<0.01
Supination (°)	55.3 (range 40~80)	58.8 (range 25~85)	0.455
Pronation (°)	50.6 (range 30~65)	53.8 (range 30~80)	0.427
Mayo score	66.6 (range 60~75)	71.3 (35~85)	0.015
MSTS score	71.6 (range 57.1~82.9)	81.7 (range 60~88.6)	<0.01
VAS score	1.2 (range 0~3)	1.3 (range 0~4)	0.985
Grip strength (% of normal side) (%)	54.8 (range 44.4~70.8)	61.6 (range 27.7~76)	<0.01
The variations before and after surgery
Extension (°)	7.2 (range -20~30)	29.7 (range 5~55)	<0.01
Flexion (°)	1.3 (rang -20~20)	12.5 (range 0~35)	0.015
Supination (°)	21.6 (range 0~50)	24.1 (range 0~50)	0.593
Pronation (°)	16.6 (range -5~40)	21.6 (range 10~40)	0.341
Mayo score	48.8 (rang 40~65)	51.6 (range 20~70)	0.047
MSTS score	18.2 (range 5.7~34.3)	27.5 (range -2.9~57.1)	0.017
VAS score	4.4 (range 2~6)	4.3 (range 1~6)	0.891
Grip strength (% of normal side) (%)	25.9 (range 11.5~48.8)	28.2 (range 9.9~50.7)	0.564

Table 5 Summary of the most important published studies on osteoarticular allograft of GCT in the distal radius

Author (year)	Patients (n)	Follow-up (months)	Grip strength	Range of motion	Functional scores	Oncologic Results	Degenerative change	complications
Richard et al[34](1977)	3	Mean 20.7 (range,6-33)	Mean 35%^a	Extension: mean 26.7° Flexion: mean 61.7° Pronation: mean 70° Supination: mean 71.7°	NA.	continuous disease free	NA	Subluxation (1)
Cheng et al[35](2001)	4	Mean 60 (range,36-96)	Mean 70.3%^a	Mean 70%^a	MSTS: excellent (3); good (1)	continuous disease free	OA^cof the radiocarpal joint (2)	Radioulnar diastasis (2); ulnar translation of carpus (2)
Bianchi et al[12](2005)	9	Mean 57.3 (range,26-145)	NA	Extension: mean 35.5° Flexion: mean 47.7°	Functional outcome (%)^a: mean 91.2	Local recurrence (3); lung metastasis (2)	All developed radiographic narrowing	Moderate pain during daily activities (1); ulnar subluxation (5)
Szabo et al[19](2006)	9	Mean 100 (range,39-219)	Mean 77%^a	Extension: mean 52° Flexion: mean 50° Pronation: mean 80° Supination: mean 67°	DASH: mean 15; SF-36: mean 72; Mayo: mean 73	continuous disease free	All developed radiographic narrowing	Minor infection (1); fixation failure (1); flexor carpi radialis tendonitis (1); stress fracture of the allograft (1); ulnar synostoses (2)
Asavamongkolkul et al[36](2009)	8	Mean 52.7 (range,40.5-90.9)	Mean 72.1%^a	Mean 72.5%^a	MSTS: mean 93%	Lung metastasis (1)	OA^cof the radiocarpal joint (2)	Nonunion (2); graft fracture (1); ulnar translation of carpus (1)
Duan et al[13](2013)	15	Mean 62.4 (range,36-139)	Mean 27 hg	Extension: mean 46.7° Flexion: mean 33.3° Pronation: mean 72.3° Supination: mean 61.3°	SF-36: mean 71; Mayo: mean 70	Soft tissues recurrence (1)	All developed radiographic narrowing	No
Rabitsch et al[33](2013)	4	Mean 32 (range,4-121)	Return to prior work	Extension/flexion: mean 60°/38°; Pronation/supination: mean 77°/77°	Mayo: mean 84; DASH: mean 8;	continuous disease free	All patients but no pain	Nonunion (1)
Li et al[37](2015)	17	Mean 84.7 (range,42-131)	Mean 63.7%^a	Extension+Flexion (%)^a: mean 43.5 Pronation+Supination (%)^a: mean 56.2	MSTS: mean 75%	Local recurrence (1)	Severe (5); moderate (9); minor (3)	Subluxation (3); nonunion (1); avulsion rupture of tendon (2); revised with another allograft (1);
Wysocki et al[4](2015)	4	Mean 245	Mean 69.4%^a	Extension: mean 40° Flexion: mean 67.5° Pronation: mean 75° Supination: mean 52.5°	DASH: mean 20; MSTS: mean 87%	continuous disease free	Grade II (2) arthrosis^d	Nonunion (1)

^a % of contralateral side; NA, not applicable; ^costeoarthritis; ^d the grading scale of Knirk and Jupiter

Table 6 Summary of the most important published studies on prosthetic replacement of GCT in the distal radius

Author (year)	Patients (n)	Follow-up (months)	Prosthesis design	Device for soft tissue restoration	Grip strength	Range of motion	Functional scores	Oncologic results	Degenerative change	complications
Gold et al[10](1957)	1	59	Cemented stem	NA	Sufficient for heavy work	A small range of motion	NA	continuous disease free	NA	Fracture of the prosthesis
Hatano et al [11](2006)	1	168	Cemented stem	purpose-made holes	71%^a	Extension: 30° Flexion: 15° Pronation: 30° Supination: 45°	Enneking scale: 83%	continuous disease free	NA	NA
Gokaraju et al [41](2009)	1	56	Cemented stem	3mm purpose-made holes	equal to the contralateral side	Extension: 40° Flexion: 20° Pronation: full Supination: 45°	Full DASH: 10.3/100	continuous disease free	Mild	ulna translation of carpus
Natarajan et al[22](2009)	16	Mean 78 (range, 24–156)	bipolar hinge component with cemented stem	NA	NA	Extension: 20° Flexion: 25° Pronation: 60° Supination: 40°	MSTS: mean 74%	NA	-	Aseptic loosening (2); wound infection (2); skin flap necrosis (2)
Damert et al[28](2013)	1	24	Cemented stem	NA	34.8%^a	Extension: 45° Flexion: 10° Pronation: 80° Supination: 10°	DASH: 25	continuous disease free	NA	No
Hariri et al[27](2013)	1	33	Cemented stem	NA	63%^a	Extension: 70° Flexion: 20° Pronation: 70° Supination: 60°	Quick DASH: 52.3/100; Enneking scale: 83%	continuous disease free	NA	No
Zhang et al[9](2015)	11	Mean 55.5 (range, 24–83)	Cemented stem	purpose-made holes	Mean 33.1%^a	Extension: mean 40.1° Flexion: mean 30° Pronation: mean 38.2° Supination: mean 46.4°	MSTS: mean 80%	continuous disease free	No	Superficial infection (1); pain (1)
Wang et al[20](2016)	10	Mean 52 (range, 24–90)	Cemented stem	purpose-made holes	Mean 68%^a	Extension: mean 40.1° Flexion: mean 30° Pronation: mean 38.2° Supination: mean 46.4°	Mayo: mean 68	continuous disease free	Grade 0 (7); grade 1 (2); grade 2 (1) arthrosis^b	Aseptic loosing (1); pain (2)

NA, not applicable; ^a % of contralateral side; ^b the grading scale of Knirk and Jupiter

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The Functional Outcomes and Complications of Different Reconstruction Methods for Giant Cell Tumor of the Distal Radius: Comparison of Osteoarticular Allograft and Three-Dimensional-Printed Prosthesis

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