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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 early. The prosthesis that we use is custom-made from


Eagle Osteon Technologies, Chennai, India. Figure 1
A giant cell tumor (GCT) of bone is a benign and locally shows the stretch by the engineers, which is made
aggressive lesion. It usually involves the metaphyseal- based on measured roentgenograms and the length of
epiphyseal region of long bones. It is a relatively rare at resection, which is determined by the surgeons. The
the ankle but is known to behave unpredictably when prosthesis is made from titanium alloy and consists of
situated at that location. It comprises less than 4% of two components that are locked together by the three
GCT cases. The usual mode of treatment for these screws and a central peg (Fig. 2a). The distal compo-
tumors is extended curettage but for recurrence of the nent has a saddle joint that sits on the dome of the
tumor, below-knee amputation has been the treatment talus and is stabilized by four screws into the talus
of choice because complete excision of tumors in this (Fig. 2b). The saddle joint consists of a bar lined with a
region is difficult to achieve and reconstruction options polyethylene liner that articulates with an oblong hole
usually give poor functional results. This is due to the at the superior aspect of the talus end of the prosthesis.
tumors subcutaneous location and the proximity of the The proximal component has a cylindrical stem that is
distal tibia to the neurovascular bundle and tendons. cemented intramedullary to the proximal end of the
However, limb salvage has replaced amputation as the tibia. The articular surfaces are lined by ultra-high
mainstay of surgical treatment in tumors in the other molecular-weight polyethylene. The movement at the
regions such as the proximal femur, the distal femur and ankle is not a hinge movement, rather a saddle joint
the proximal tibia, principally because of improvements movement to better mimic the mechanics of a normal
in surgical technique and in the design and production ankle joint.
of the implant.
An endoprosthetic replacement enables the early res-
toration of function as the patients can bear weight Operative technique
The anterior lateral approach was used for all patients.
The biopsy tract is excised in all patients. The tumor is
Correspondence: Dr Vivek Ajit Singh FRCS MS(Ortho), dissected with wide margins. The proximal bone cuts
Department of Orthopedics, University of Malaya, Kuala were made 3 cm from the distal margin of the tumor.
Lumpur 50603, Malaysia. Email: drvivek69@gmail.com The cartilage over the talar dome is shaved off and the
Accepted for publication 28 March 2012. head of talus is fashioned to fit the cup end of the

2012 Blackwell Publishing Asia Pty Ltd


2 V Ajit Singh et al.

7
7
120

120
9
9
18
18
100

115

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 To date there are only three publications on the endo-
and secured with four fully treaded 4-mm cancellous prosthetic replacement of the distal tibia.15 We report
screws (Fig. 3a). The proximal end of the bone is reamed our series of four cases of recurrent GCT of the distal
to 2 mm bigger than the stem diameter and the stem is tibia that were successfully treated with wide resection
cemented in without a cement spacer. The two compo- and endoprosthetic reconstruction. We believe that
nents are reduced and locked together by the peg and distal tibial endoprosthetic replacement is a viable
secured by three screws (Fig. 3b). The final implant in option that enables patients to maintain their limbs and
situ is shown in Figure 4. function.

2012 Blackwell Publishing Asia Pty Ltd Asia-Pac J Clin Oncol 2012
Endoprosthesis of distal tibia 3

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 taken 30 months after the surgery is shown in Figure 6.


There is no evidence of loosening. His latest Muscu-
Patient 1 loskeletal Tumor Society (MSTS) score is 86.7%.
This is a 25-year old man who presented with a right
ankle swelling 8 years prior to presentation. A diagnosis Case 2
of GCT of the distal tibia was made by radiography The second patient is a 16-year-old girl who presented
(Fig. 5a) and confirmed by a needle biopsy. He under- with a 6-month history of aching pain and swelling of
went an extended curettage and bone grafting. No adju- the right ankle when she was first seen, by our oncology
vant treatment was given. In August 2007 he presented unit in 2004. The patient had no systemic symptoms or
again with recurrence of pain over the ankle for over 6 problems related to the joint. On examination, the
months. The pain had been getting progressively worse patient walked normally and had slight tenderness to
and a roentgenogram showed a recurrent expansile palpation of the distal part of the tibia. Radiographs
lesion over the distal tibia, suggestive of a recurrence, revealed a lytic expansile lesion in the distal metaphysis
Campanacci grade 2 (Fig. 5b). The diagnosis was recon- of the right tibia. The diagnosis of a GCT of the bone
firmed with a needle biopsy. A wide resection of the was established with a biopsy. The lesion was curetted,
distal tibia with endoprosthesis replacement was done the distal end of fibular was excised together with the
on September 2007. The patient was allowed full weight lesion and the cavity was filled with corticocancellous
bearing 3 days after the surgery and started walking bone graft from the iliac crest and the middle portion
unaided 2 weeks afterwards. The latest radiograph of fibula was used to reconstruct the ankle joint. The

Asia-Pac J Clin Oncol 2012 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
Endoprosthesis of distal tibia 5

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 ing motion in the right ankle when our unit first saw him
within 3 months. in March 2004. Radiographs revealed a lytic expansile
She remained asymptomatic for 3 years. In Decem- lesion in the distal part of the right tibia. After a biopsy
ber 2007 she noted progressively worsening pain with confirmation, an extended curettage and autologous
activity and the recurrent swelling of the right ankle. bone grafting was carried out from the iliac crest. In
Radiographs revealed a recurrence of the lytic lesion in September 2007 the patient had recurrence of disease.
the distal part of the right tibia, as shown in Figure 7a. He went to another institution and an amputation was
A study of a specimen obtained by means of a needle planned but he defaulted.
biopsy revealed the diagnosis of recurrent GCT of the In February 2008, when he came to see us, he was
bone. A wide resection of distal tibia with endopros- walking with non-weight bearing crutches. Radiographs
thesis reconstruction was done in February 2008. Post- show the expansion and thinning of the distal tibia
operatively, she recovered well and went back to cortex, suggestive of a recurrence of the tumor, Cam-
college. About 2 years after the surgery, she developed panacci grade 3 (Fig. 8a). After a biopsy confirmation, a
over the shin a sinus discharging pus and complained wide resection and endoprosthesis replacement was
of occasional mechanical pain over the region. The done. He was back to function within 2 weeks. On
radiographs taken show septic loosening over the follow up, it was noted that his ankle was in 15 degrees
proximal end of the implant (Fig. 7b). The wound cul- valgus and the follow-up radiograph showed talar col-
tures grew a methicillin-resistant staphylococcus infec- lapse (Fig. 8b) but the implant was still stable and he is
tion. She refused two-stage revision and is currently on able to bear his full weight without any pain. His latest
chronic antibiotic suppression. Her latest MSTS score MSTS score is 80%.
is 73.3%.
Case 4
Case 3 This is a 26-year-old man who presented with a
The third patient is a 27-year-old man who presented 6-month history of right distal tibia swelling and a
with a 9-month history of increasing pain and decreas- 2-month history of pain. The radiographs and magnetic

Asia-Pac J Clin Oncol 2012 2012 Blackwell Publishing Asia Pty Ltd
6 V Ajit Singh et al.

a b

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.

Figure 9 (a) MRI of the right ankle


with expansion of the distal tibia with
a b tumor, (b) resected specimen with the
custom made endoprosthesis.

resonance imaging at presentation are shown in A summary of all the four patients is given in Table 1.
Figure 9a. We opted for an endoprosthesis replacement The details of the MSTS score are shown in Table 2. The
as the lesion was extensive and involved almost the average MSTS score 82.5%.
entire distal tibia. He underwent the surgery in July
2009. The resected specimen and the endoprosthesis are
DISCUSSION
shown in Figure 9b. He was bearing weight on day 2
postoperatively and was able to walk without assistance GCT generally occurs in those with mature skeletons
after 2 weeks. His latest MSTS score is 90%. with a peak incidence in the third decade of life. GCT of

2012 Blackwell Publishing Asia Pty Ltd Asia-Pac J Clin Oncol 2012
Endoprosthesis of distal tibia 7

Table 1 Summary of the four patients with giant cell tumor (GCT)
(a)
Duration of follow up
Patient no Age Sex Side of lesion Diagnosis (months) MSTS score Complication
1. 25 Male Right Recurrent GCT 36 86.7 None
2. 16 Female Right Recurrent GCT 32 73.3 Deep infection
3. 27 Male Right Recurrent GCT 30 80% Talar collapse
4. 26 Male Right GCT 12 90% None
MSTS, Musculoskeletal Tumor Society score.

Table 2 Details of the functional results (MSTS Scores)


Functional Emotional External Walking Total functional
Patient no Pain activities acceptance support ability Gait score (%)
1 4 3 4 5 5 5 86.7
2 4 4 3 4 4 3 73.3
3 4 4 3 5 4 4 80
4 5 4 5 5 5 3 90

the foot and ankle are very rare and occur commonly in series of six cases of osteosarcoma of the distal tibia
young adults with an average age of 28.8 years.6 treated with fibular grafts (vascularized and non-
Various forms of treatment for GCT of the distal tibia vascularized) stabilized with an Ilizarov external fixator.
have been proposed, including extended intra-lesional It took an average of 13.2 months to achieve union and
curettage, with the use of phenol, cryotherapy and burr he reported that the vascularized grafts gave a better
and bone grafting, cryotherapy, cementation, limb union than non-vascularized grafts. Their average MSTS
salvage and even amputation. Traditionally, transtibial score was 70%. Jeon et al.11 reported a series of nine
amputation has been the treatment of choice for treating patients where the reconstruction was done with pas-
local bone tumors involving the distal tibia and fibula.7 teurized autograft. In his series, six patients had post-
A wide margin of resection and a satisfactory functional operative complications such as a superficial wound
result with an appropriate below-knee prosthesis can be infection, non-union and fracture.
achieved with amputation. However, amputation is In our series of four cases, patients were allowed to
associated with significant psychological, physical, bear weight 2 to 3 days after surgery and after 2 weeks
social and financial cost to patients. were back to full function. The average MSTS score in
As the surgery of limb salvage has developed, recon- this series is 82.5%. We did not encounter soft tissue
struction of ankle joint has been attempted using problems, as all four cases were GCT and the resections
autografts, allografts and endoprosthesis. Among the were done close to the bone, preserving the soft tissue
many options of reconstruction, arthrodesis was envelope. Thus, limb salvage operations using distal end
regarded as the best option. It provided excellent stabil- endoprosthesis have potential advantages compared to
ity of the ankle joint and avoided problems relating to biological alternatives. These include early stability,
prosthetic implantation. A study by Casadei et al.8 early mobilization and weight bearing, and a better
reported a good functional outcome in patients with appearance and psychological acceptance. All the
distal tibia tumor treated by resection and autogenous patients in our series showed excellent stability of their
bone graft arthrodesis. However, arthrodesis of the ankles at the latest follow up. All the patients had an
ankle has several limitations, including the loss of joint excellent oncology outcome. One had a talar collapse
movement, a long period of recovery, the need for mul- without loosening of the implant or compromising his
tiple operations to achieve arthrodesis and the possibil- function. Another patient developed deep infection with
ity of non-union. This was overcome by the use of methicillin-resistant staphylococcus but still maintained
vascularized free fibular graft, which gave better union the endoprosthesis with chronic antibiotic suppression.
rates but still a stiff ankle.9 Shalaby et al.10 reported a The remaining two patients are functioning well. Abudu

Asia-Pac J Clin Oncol 2012 2012 Blackwell Publishing Asia Pty Ltd
8 V Ajit Singh et al.

et al.12 concluded in their series of five patients that following tumour excision. J Bone Joint Surg Br 1995;
endoprosthesis replacement of the distal tibia has a sig- 77-B (Suppl 3): 330.
nificant medium-term morbidity and functional deterio- 3 Lee SH, Kim H-S, Park Y-B, Rhie T-Y, Lee HK.Prosthetic
ration. Shekkeris et al.13 in his series of six patients, reconstruction for tumours of the distal tibia and fibula.
J Bone Joint Surg Br 1999; 81-B: 8037.
reported deep infection in two cases that eventually lead
4 Natarajan MV, Annamalai K, Williams S, Selvaraj R,
to amputation. The average MSTS score for the remain-
Rajagopal TS. Limb salvage in distal tibial osteosarcoma
ing four patients in his series was 70%. He concluded using a custom mega prosthesis. Int Orthop 2000; 24:
that endoprosthetic replacement is a viable option pro- 2824.
viding that patients understand the risk of uncontrolled 5 Hamada K, Naka N, Murata Y, Yasui Y, Joyama S, Araki
infection, which may lead to amputation. N. Prosthetic reconstruction for tumors of the distal tibia.
The prosthesis that we use is custom made by Eagle Report of two cases. Foot (Edinb) 2011; 21: 15761.
Osteon Technologies, Chennai, India. It has a semi- 6 Kamath S. Giant cell tumour around the foot and ankle.
constrained joint at the ankle portion. Our design does Foot Ankle Surg 2006; 12: 99102.
not have an osseous integration collar proximally for 7 Ozaki T, Hillman A, Lindner N, Winkelmann W. Surgical
long-term bone in growth and stability. We foresee loos- treatment of bone sarcomas of the fibula: analysis of 19
cases. Arch Orthop Trauma Surg 1997; 116: 4759.
ening as one of the problems that we might face in the
8 Casadei R, Ruggieri P, Giuseppe T, Biagini R, Mercuri M.
long term due to the high torsional forces that are trans-
Ankle resection arthrodesis in patients with bone tumours.
mitted through the boneimplant junction. We have Foot Ankle Int 1994; 15: 2429.
highlighted this potential problem to the manufacturers 9 Bishop AT, Wood MB, Sheetz KK. Arthrodesis of the ankle
and have advised them to incorporate it in their future with a free vascularised autogenous bone graft: reconstruc-
designs. So far, in our follow up, there have been no tion of segmental loss of bone secondary to osteomyelitis,
cases of loosening. tumor or trauma. J Bone Joint Surg Am 1995; 77-A: 1867
We believe that distal endoprosthestic replacement is 75.
a feasible option in cases of extensive distal tibia tumors 10 Shalaby S, Salaby H, Bassiony A. Limb salvage for oste-
(especially locally aggressive tumors) that would other- osarcoma of the distal tibia with resection arthrodesis,
wise require amputation. It enables us to maintain a autogenous fibular graft and ilizarov external fixator.
J Bone Joint Surg Br 2006; 88-B: 16426.
functional ankle joint and an almost full function of the
11 Jeon D-G, Kim MS, Cho WH, Song WS, Lee S-Y. Recon-
limb. However, it is still early to predict the long-term
struction with pasteurized autograft for distal tibial tumor.
survival of this implant in our series of patients. Arch Orthop Trauma Surg 2008; 128: 15965.
12 Abudu A, Grimer RJ, Tilman RM, Carter SR. Endo-
REFERENCES prosthetic replacement of the distal tibia and ankle
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tibia replacement. In: limb salvage current trends. Proceed- 13 Shekkeris AS, Hanna SA, Sewell MD et al. Endoprosthetic
ings of the 7th ISOLS Meeting, 1993; 562. reconstruction of the distal tibia and ankle joint after resec-
2 Abudu A, Tillman RM, Carter SR, Grimer RJ. Endopros- tion of primary bone tumours. J Bone Joint Surg Br 2009;
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2012 Blackwell Publishing Asia Pty Ltd Asia-Pac J Clin Oncol 2012

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