bs_bs_banner
Asia-Pacific Journal of Clinical Oncology 2012
doi: 10.1111/j.1743-7563.2012.01553.x
CASE REPORT
Endoprosthetic reconstruction for giant cell tumors of the
distal tibia: A short term review
Vivek AJIT SINGH,1 Norzatulsyima NASIRUDIN2 and Michael BERNATT2
1
Department of Orthopedics, University of Malaya and 2Department of Orthopedics, Ministry of Health, Kuala Lumpur,
Malaysia
Abstract
Custom-made endoprosthetic reconstruction for distal tibia tumors is a viable option of treatment in
carefully selected patients. It maintains satisfactory function and provides good pain relief. We report four
cases of giant cell tumors of the distal tibia successfully treated by endoprosthetic reconstruction. This is a
feasible option in cases of this nature and offers a better function than the other available options.
Key words: pathology, sarcoma, surgical oncology.
INTRODUCTION
A giant cell tumor (GCT) of bone is a benign and locally
aggressive lesion. It usually involves the metaphysealepiphyseal region of long bones. It is a relatively rare at
the ankle but is known to behave unpredictably when
situated at that location. It comprises less than 4% of
GCT cases. The usual mode of treatment for these
tumors is extended curettage but for recurrence of the
tumor, below-knee amputation has been the treatment
of choice because complete excision of tumors in this
region is difficult to achieve and reconstruction options
usually give poor functional results. This is due to the
tumor’s subcutaneous location and the proximity of the
distal tibia to the neurovascular bundle and tendons.
However, limb salvage has replaced amputation as the
mainstay of surgical treatment in tumors in the other
regions such as the proximal femur, the distal femur and
the proximal tibia, principally because of improvements
in surgical technique and in the design and production
of the implant.
An endoprosthetic replacement enables the early restoration of function as the patients can bear weight
Correspondence: Dr Vivek Ajit Singh FRCS MS(Ortho),
Department of Orthopedics, University of Malaya, Kuala
Lumpur 50603, Malaysia. Email: [email protected]
Accepted for publication 28 March 2012.
© 2012 Blackwell Publishing Asia Pty Ltd
early. The prosthesis that we use is custom-made from
Eagle Osteon Technologies, Chennai, India. Figure 1
shows the stretch by the engineers, which is made
based on measured roentgenograms and the length of
resection, which is determined by the surgeons. The
prosthesis is made from titanium alloy and consists of
two components that are locked together by the three
screws and a central peg (Fig. 2a). The distal component has a saddle joint that sits on the dome of the
talus and is stabilized by four screws into the talus
(Fig. 2b). The saddle joint consists of a bar lined with a
polyethylene liner that articulates with an oblong hole
at the superior aspect of the talus end of the prosthesis.
The proximal component has a cylindrical stem that is
cemented intramedullary to the proximal end of the
tibia. The articular surfaces are lined by ultra-high
molecular-weight polyethylene. The movement at the
ankle is not a hinge movement, rather a saddle joint
movement to better mimic the mechanics of a normal
ankle joint.
Operative technique
The anterior lateral approach was used for all patients.
The biopsy tract is excised in all patients. The tumor is
dissected with wide margins. The proximal bone cuts
were made 3 cm from the distal margin of the tumor.
The cartilage over the talar dome is shaved off and the
head of talus is fashioned to fit the cup end of the
2
V Ajit Singh et al.
7
7
120
120
9
115
100
18
9
18
Figure 1 The planned stretch by the engineers based on measured roentgenograms shown on the left.
a
b
Figure 2 (a) The two components of the prosthesis connected together by locking mechanism and three screws, (b) the distal
portion of the prosthesis that sits on the dome of the talus; stabilized by four screws.
prosthesis. This component is cemented onto the talus
and secured with four fully treaded 4-mm cancellous
screws (Fig. 3a). The proximal end of the bone is reamed
to 2 mm bigger than the stem diameter and the stem is
cemented in without a cement spacer. The two components are reduced and locked together by the peg and
secured by three screws (Fig. 3b). The final implant in
situ is shown in Figure 4.
© 2012 Blackwell Publishing Asia Pty Ltd
To date there are only three publications on the endoprosthetic replacement of the distal tibia.1–5 We report
our series of four cases of recurrent GCT of the distal
tibia that were successfully treated with wide resection
and endoprosthetic reconstruction. We believe that
distal tibial endoprosthetic replacement is a viable
option that enables patients to maintain their limbs and
function.
Asia-Pac J Clin Oncol 2012
3
Endoprosthesis of distal tibia
a
b
Figure 3 (a) The distal component cemented onto the talar dome and secured in with four screws, (b) the proximal and distal
components are reduced and locked together with the peg and three screws.
Figure 4 Intraoperative image of
the implant in situ. Proximally the
implant is cemented into the proximal
tibia and distally it is cemented into
the dome of talus, augmented with
screw fixation (two screws anteriorly
and two screws posteriorly).
CASE REPORTS
Patient 1
This is a 25-year old man who presented with a right
ankle swelling 8 years prior to presentation. A diagnosis
of GCT of the distal tibia was made by radiography
(Fig. 5a) and confirmed by a needle biopsy. He underwent an extended curettage and bone grafting. No adjuvant treatment was given. In August 2007 he presented
again with recurrence of pain over the ankle for over 6
months. The pain had been getting progressively worse
and a roentgenogram showed a recurrent expansile
lesion over the distal tibia, suggestive of a recurrence,
Campanacci grade 2 (Fig. 5b). The diagnosis was reconfirmed with a needle biopsy. A wide resection of the
distal tibia with endoprosthesis replacement was done
on September 2007. The patient was allowed full weight
bearing 3 days after the surgery and started walking
unaided 2 weeks afterwards. The latest radiograph
Asia-Pac J Clin Oncol 2012
taken 30 months after the surgery is shown in Figure 6.
There is no evidence of loosening. His latest Musculoskeletal Tumor Society (MSTS) score is 86.7%.
Case 2
The second patient is a 16-year-old girl who presented
with a 6-month history of aching pain and swelling of
the right ankle when she was first seen, by our oncology
unit in 2004. The patient had no systemic symptoms or
problems related to the joint. On examination, the
patient walked normally and had slight tenderness to
palpation of the distal part of the tibia. Radiographs
revealed a lytic expansile lesion in the distal metaphysis
of the right tibia. The diagnosis of a GCT of the bone
was established with a biopsy. The lesion was curetted,
the distal end of fibular was excised together with the
lesion and the cavity was filled with corticocancellous
bone graft from the iliac crest and the middle portion
of fibula was used to reconstruct the ankle joint. The
© 2012 Blackwell Publishing Asia Pty Ltd
4
V Ajit Singh et al.
a
b
Figure 5 (a) Roentgenograms of the right distal tibia showing an expansile lesion in the right distal tibia located at the metaphysis
extending into the epiphysis, (b) the repeat roentgenograms after 8 years, showing recurrence of the expansile lesion of the distal
tibia with multiple areas of cavitation.
Figure 6 The latest radiograph for patient 1. There is no evidence of loosening and there is bone growth over the proximal
attachment of prosthesis.
© 2012 Blackwell Publishing Asia Pty Ltd
Asia-Pac J Clin Oncol 2012
5
Endoprosthesis of distal tibia
a
b
Figure 7 (a) The anterior posterior views of the right distal tibia. There is a lytic lesion on the medial aspect of the right distal tibia
extending into the distal fibular. There is erosion of the lateral fibular cortex, (b) loosening over the proximal end of the implant.
postoperative patient resumed full weight-bearing
within 3 months.
She remained asymptomatic for 3 years. In December 2007 she noted progressively worsening pain with
activity and the recurrent swelling of the right ankle.
Radiographs revealed a recurrence of the lytic lesion in
the distal part of the right tibia, as shown in Figure 7a.
A study of a specimen obtained by means of a needle
biopsy revealed the diagnosis of recurrent GCT of the
bone. A wide resection of distal tibia with endoprosthesis reconstruction was done in February 2008. Postoperatively, she recovered well and went back to
college. About 2 years after the surgery, she developed
over the shin a sinus discharging pus and complained
of occasional mechanical pain over the region. The
radiographs taken show septic loosening over the
proximal end of the implant (Fig. 7b). The wound cultures grew a methicillin-resistant staphylococcus infection. She refused two-stage revision and is currently on
chronic antibiotic suppression. Her latest MSTS score
is 73.3%.
Case 3
The third patient is a 27-year-old man who presented
with a 9-month history of increasing pain and decreas-
Asia-Pac J Clin Oncol 2012
ing motion in the right ankle when our unit first saw him
in March 2004. Radiographs revealed a lytic expansile
lesion in the distal part of the right tibia. After a biopsy
confirmation, an extended curettage and autologous
bone grafting was carried out from the iliac crest. In
September 2007 the patient had recurrence of disease.
He went to another institution and an amputation was
planned but he defaulted.
In February 2008, when he came to see us, he was
walking with non-weight bearing crutches. Radiographs
show the expansion and thinning of the distal tibia
cortex, suggestive of a recurrence of the tumor, Campanacci grade 3 (Fig. 8a). After a biopsy confirmation, a
wide resection and endoprosthesis replacement was
done. He was back to function within 2 weeks. On
follow up, it was noted that his ankle was in 15 degrees
valgus and the follow-up radiograph showed talar collapse (Fig. 8b) but the implant was still stable and he is
able to bear his full weight without any pain. His latest
MSTS score is 80%.
Case 4
This is a 26-year-old man who presented with a
6-month history of right distal tibia swelling and a
2-month history of pain. The radiographs and magnetic
© 2012 Blackwell Publishing Asia Pty Ltd
6
V Ajit Singh et al.
b
a
Figure 8 (a) Expansile lesion over the right distal tibia (Campanacci grade 3), (b) talar collapse with backing out of screws but the
implant appears stable.
a
Figure 9 (a) MRI of the right ankle
with expansion of the distal tibia with
tumor, (b) resected specimen with the
custom made endoprosthesis.
b
resonance imaging at presentation are shown in
Figure 9a. We opted for an endoprosthesis replacement
as the lesion was extensive and involved almost the
entire distal tibia. He underwent the surgery in July
2009. The resected specimen and the endoprosthesis are
shown in Figure 9b. He was bearing weight on day 2
postoperatively and was able to walk without assistance
after 2 weeks. His latest MSTS score is 90%.
© 2012 Blackwell Publishing Asia Pty Ltd
A summary of all the four patients is given in Table 1.
The details of the MSTS score are shown in Table 2. The
average MSTS score 82.5%.
DISCUSSION
GCT generally occurs in those with mature skeletons
with a peak incidence in the third decade of life. GCT of
Asia-Pac J Clin Oncol 2012
7
Endoprosthesis of distal tibia
Table 1 Summary of the four patients with giant cell tumor (GCT)
(a)
Patient no
Age
Sex
1.
2.
3.
4.
25
16
27
26
Male
Female
Male
Male
Side of lesion
Right
Right
Right
Right
Diagnosis
Duration of follow up
(months)
MSTS score
Complication
Recurrent GCT
Recurrent GCT
Recurrent GCT
GCT
36
32
30
12
86.7
73.3
80%
90%
None
Deep infection
Talar collapse
None
MSTS, Musculoskeletal Tumor Society score.
Table 2 Details of the functional results (MSTS Scores)
Patient no
1
2
3
4
Pain
Functional
activities
Emotional
acceptance
External
support
Walking
ability
Gait
Total functional
score (%)
4
4
4
5
3
4
4
4
4
3
3
5
5
4
5
5
5
4
4
5
5
3
4
3
86.7
73.3
80
90
the foot and ankle are very rare and occur commonly in
young adults with an average age of 28.8 years.6
Various forms of treatment for GCT of the distal tibia
have been proposed, including extended intra-lesional
curettage, with the use of phenol, cryotherapy and burr
and bone grafting, cryotherapy, cementation, limb
salvage and even amputation. Traditionally, transtibial
amputation has been the treatment of choice for treating
local bone tumors involving the distal tibia and fibula.7
A wide margin of resection and a satisfactory functional
result with an appropriate below-knee prosthesis can be
achieved with amputation. However, amputation is
associated with significant psychological, physical,
social and financial cost to patients.
As the surgery of limb salvage has developed, reconstruction of ankle joint has been attempted using
autografts, allografts and endoprosthesis. Among the
many options of reconstruction, arthrodesis was
regarded as the best option. It provided excellent stability of the ankle joint and avoided problems relating to
prosthetic implantation. A study by Casadei et al.8
reported a good functional outcome in patients with
distal tibia tumor treated by resection and autogenous
bone graft arthrodesis. However, arthrodesis of the
ankle has several limitations, including the loss of joint
movement, a long period of recovery, the need for multiple operations to achieve arthrodesis and the possibility of non-union. This was overcome by the use of
vascularized free fibular graft, which gave better union
rates but still a stiff ankle.9 Shalaby et al.10 reported a
Asia-Pac J Clin Oncol 2012
series of six cases of osteosarcoma of the distal tibia
treated with fibular grafts (vascularized and nonvascularized) stabilized with an Ilizarov external fixator.
It took an average of 13.2 months to achieve union and
he reported that the vascularized grafts gave a better
union than non-vascularized grafts. Their average MSTS
score was 70%. Jeon et al.11 reported a series of nine
patients where the reconstruction was done with pasteurized autograft. In his series, six patients had postoperative complications such as a superficial wound
infection, non-union and fracture.
In our series of four cases, patients were allowed to
bear weight 2 to 3 days after surgery and after 2 weeks
were back to full function. The average MSTS score in
this series is 82.5%. We did not encounter soft tissue
problems, as all four cases were GCT and the resections
were done close to the bone, preserving the soft tissue
envelope. Thus, limb salvage operations using distal end
endoprosthesis have potential advantages compared to
biological alternatives. These include early stability,
early mobilization and weight bearing, and a better
appearance and psychological acceptance. All the
patients in our series showed excellent stability of their
ankles at the latest follow up. All the patients had an
excellent oncology outcome. One had a talar collapse
without loosening of the implant or compromising his
function. Another patient developed deep infection with
methicillin-resistant staphylococcus but still maintained
the endoprosthesis with chronic antibiotic suppression.
The remaining two patients are functioning well. Abudu
© 2012 Blackwell Publishing Asia Pty Ltd
8
et al.12 concluded in their series of five patients that
endoprosthesis replacement of the distal tibia has a significant medium-term morbidity and functional deterioration. Shekkeris et al.13 in his series of six patients,
reported deep infection in two cases that eventually lead
to amputation. The average MSTS score for the remaining four patients in his series was 70%. He concluded
that endoprosthetic replacement is a viable option providing that patients understand the risk of uncontrolled
infection, which may lead to amputation.
The prosthesis that we use is custom made by Eagle
Osteon Technologies, Chennai, India. It has a semiconstrained joint at the ankle portion. Our design does
not have an osseous integration collar proximally for
long-term bone in growth and stability. We foresee loosening as one of the problems that we might face in the
long term due to the high torsional forces that are transmitted through the bone–implant junction. We have
highlighted this potential problem to the manufacturers
and have advised them to incorporate it in their future
designs. So far, in our follow up, there have been no
cases of loosening.
We believe that distal endoprosthestic replacement is
a feasible option in cases of extensive distal tibia tumors
(especially locally aggressive tumors) that would otherwise require amputation. It enables us to maintain a
functional ankle joint and an almost full function of the
limb. However, it is still early to predict the long-term
survival of this implant in our series of patients.
V Ajit Singh et al.
3
4
5
6
7
8
9
10
11
12
REFERENCES
1 Unwin P, Cannon S, Kemp H et al. The Stanmore distal
tibia replacement. In: limb salvage current trends. Proceedings of the 7th ISOLS Meeting, 1993; 562.
2 Abudu A, Tillman RM, Carter SR, Grimer RJ. Endoprosthesic reconstruction of the distal tibia and ankle joint
© 2012 Blackwell Publishing Asia Pty Ltd
13
following tumour excision. J Bone Joint Surg Br 1995;
77-B (Suppl 3): 330.
Lee SH, Kim H-S, Park Y-B, Rhie T-Y, Lee HK.Prosthetic
reconstruction for tumours of the distal tibia and fibula.
J Bone Joint Surg Br 1999; 81-B: 803–7.
Natarajan MV, Annamalai K, Williams S, Selvaraj R,
Rajagopal TS. Limb salvage in distal tibial osteosarcoma
using a custom mega prosthesis. Int Orthop 2000; 24:
282–4.
Hamada K, Naka N, Murata Y, Yasui Y, Joyama S, Araki
N. Prosthetic reconstruction for tumors of the distal tibia.
Report of two cases. Foot (Edinb) 2011; 21: 157–61.
Kamath S. Giant cell tumour around the foot and ankle.
Foot Ankle Surg 2006; 12: 99–102.
Ozaki T, Hillman A, Lindner N, Winkelmann W. Surgical
treatment of bone sarcomas of the fibula: analysis of 19
cases. Arch Orthop Trauma Surg 1997; 116: 475–9.
Casadei R, Ruggieri P, Giuseppe T, Biagini R, Mercuri M.
Ankle resection arthrodesis in patients with bone tumours.
Foot Ankle Int 1994; 15: 242–9.
Bishop AT, Wood MB, Sheetz KK. Arthrodesis of the ankle
with a free vascularised autogenous bone graft: reconstruction of segmental loss of bone secondary to osteomyelitis,
tumor or trauma. J Bone Joint Surg Am 1995; 77-A: 1867–
75.
Shalaby S, Salaby H, Bassiony A. Limb salvage for osteosarcoma of the distal tibia with resection arthrodesis,
autogenous fibular graft and ilizarov external fixator.
J Bone Joint Surg Br 2006; 88-B: 1642–6.
Jeon D-G, Kim MS, Cho WH, Song WS, Lee S-Y. Reconstruction with pasteurized autograft for distal tibial tumor.
Arch Orthop Trauma Surg 2008; 128: 159–65.
Abudu A, Grimer RJ, Tilman RM, Carter SR. Endoprosthetic replacement of the distal tibia and ankle
joint for aggressive bone tumours. Int Orthop 1999; 23:
291–4.
Shekkeris AS, Hanna SA, Sewell MD et al. Endoprosthetic
reconstruction of the distal tibia and ankle joint after resection of primary bone tumours. J Bone Joint Surg Br 2009;
91-B: 1378–82.
Asia-Pac J Clin Oncol 2012