Plate-assisted bone segment transport: Novel application on distal tibia defect after tumour resection. A case report

Abstract

Introduction
Reconstruction of a large bone defect of the distal tibia after limb salvage surgery is difficult. The options include custom-made ankle endoprosthetic replacement, arthrodesis, and biological or metallic intercalary reconstructions. This report introduces a technique that provides the patient with a long-lasting biological reconstruction while preserving the native ankle.

Presentation of case
We present the case of a 47-year-old man with osteosarcoma of the distal tibia. After neoadjuvant chemotherapy, wide excision was performed while preserving the ankle joint. Bone reconstruction by Plate-assisted bone segment transport (PABST) was performed with a non-invasive growing intramedullary nail. At 34 months of follow-up, there was solid union and the Musculoskeletal Tumour Society Score was 26/30.

Discussion
This is the first report of PABST after distal tibia tumour resection. It shows that this is a viable and safe method of reconstruction. Despite the use of adjuvant chemotherapy, regenerate was formed and union was achieved.

Conclusion
PABST is a useful tool in the armamentarium to tackle difficult large bone defects.

Full text

International Journal of Surgery Case Reports 84 (2021) 106079
Available online 9 June 2021
2210-2612/© 2021 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
Case report
Plate-assisted bone segment transport: Novel application on distal tibia
defect after tumour resection. A case report
Ahmed A. Eldesouqi
a
, Raymond Ching Hin Yau
b
,
*
, Wai-Yip Kenneth Ho
b
, Ying-Lee Lam
b
a
Department of Orthopaedic Surgery and Traumatology, Alexandria University, Egypt
b
Department of Orthopaedics & Traumatology, Queen Mary Hospital, Hong Kong
ARTICLE INFO
Keywords:
Tibia
Bone defect
Distraction osteogenesis
Plate-assisted bone segment transport
Case report
Precice
ABSTRACT
Introduction: Reconstruction of a large bone defect of the distal tibia after limb salvage surgery is difcult. The
options include custom-made ankle endoprosthetic replacement, arthrodesis, and biological or metallic inter-
calary reconstructions. This report introduces a technique that provides the patient with a long-lasting biological
reconstruction while preserving the native ankle.
Presentation of case: We present the case of a 47-year-old man with osteosarcoma of the distal tibia. After neo-
adjuvant chemotherapy, wide excision was performed while preserving the ankle joint. Bone reconstruction by
Plate-assisted bone segment transport (PABST) was performed with a non-invasive growing intramedullary nail.
At 34 months of follow-up, there was solid union and the Musculoskeletal Tumour Society Score was 26/30.
Discussion: This is the rst report of PABST after distal tibia tumour resection. It shows that this is a viable and
safe method of reconstruction. Despite the use of adjuvant chemotherapy, regenerate was formed and union was
achieved.
Conclusion: PABST is a useful tool in the armamentarium to tackle difcult large bone defects.
1. Introduction
Primary bone tumors affecting the distal tibial is uncommon. Only
3.8% of osteosarcomas present at the distal tibia; the 10-year survival
rate is 78% [1]. Below knee amputation was the standard treatment for
such cases. However, limb salvage surgery is now the achievable goal in
most cases in major tumour centres [2].
Reconstruction of the bony defect after limb salvage surgery is
difcult in this region. If the ankle joint cannot be salvaged, the options
include a custom-made ankle endoprosthetic replacement and arthrod-
esis. If the ankle joint can be preserved, then the options include bio-
logical (allograft, masquelet technique, recycled-bone autograft,
vascularized or non-vascularized autograft, distraction osteogenesis)
and metallic (intercalary prosthesis) reconstructions [2–4].
We report a case with distal tibia defect after osteosarcoma resection
that used Plate-assisted bone segment transport (PABST) with a non-
invasive growing intramedullary nail for reconstruction. This is the
rst report in the literature using this technique for reconstruction of the
distal tibia defect after oncological resection.
This report has been written in compliance with the SCARE 2020
guidelines [5].
2. Presentation of case
A 47-year-old non-smoking gentleman presented with right leg pain
for a few months. Examination revealed a bony hard swelling over the
anterolateral aspect of distal tibia with mild tenderness.
Radiographs revealed an eccentric lytic lesion at the distal tibial
metaphysis with cortical erosion (Fig. 1). Contrast MRI found a 27 ×44
×59 mm intramedullary mass with breach of the lateral tibial cortex
and lobulated extra-osseous component. The tumour involved the
interosseous membrane and abutted onto the bula, anterior tibial
neurovascular bundle and peroneal vessels. The lesion was T1-weighted
intermediate signal, T2-weighted high signal and contrast-enhancing
(Fig. 2). The lesion was biopsied and shown to be a chondroblastic os-
teosarcoma (Fig. 3). The patient was Enneking Stage IIB [6] as systemic
involvement was found to be negative. Two cycles of neoadjuvant
chemotherapy (Cisplatin & Doxorubicin) were given.
The surgical options of limb salvage surgery and below-knee
amputation were discussed with the patient. He refused amputation as
* Corresponding author.
E-mail address: raymondyau@ortho.hku.hk (R.C.H. Yau).
Contents lists available at ScienceDirect
International Journal of Surgery Case Reports
journal homepage: www.elsevier.com/locate/ijscr
https://doi.org/10.1016/j.ijscr.2021.106079
Received 21 April 2021; Received in revised form 26 May 2021; Accepted 3 June 2021

International Journal of Surgery Case Reports 84 (2021) 106079
2
long as wide local excision was possible. Reconstruction options were
also discussed in length during multiple visits before the surgery.
Detailed pre-operative planning was performed based on MRI and CT
images. Wide local excision of the distal tibia was performed by the
corresponding author (RY) with the aid of a custom 3D-printed cutting-
guide and computer navigation. The medial cortex of the bula was
resected together with the tumour.
The resultant bony defect measured 10 cm in length with 2 cm of
tibial plafond remaining. A Nuvasive Specialized Orthopedics (San
Diego, USA) Precice® growing nail was inserted. Corticotomy was made
Fig. 1. AP (A) & Lateral (B) radiograph of right distal tibia with lytic lesion at the lateral cortex.
Fig. 2. Contrast MRI of the tumour. T1-weighted coronal (A), sagittal (B) and T2-weighted axial (C) cuts showing cortical breach of the lateral tibial cortex and extra-
osseous soft tissue component abutting on the bula.
A.A. Eldesouqi et al.

International Journal of Surgery Case Reports 84 (2021) 106079
3
in the proximal tibia with bone gap of 8 mm. The tibial defect was
bridged with a 3.5 mm locking plate. There was an iatrogenic fracture of
the distal bula and it was xed with a distal bula locking plate (Fig. 4).
Bone transport began 17 days after the operation at 1 mm per day.
Final histology report conrmed a chondroblastic osteosarcoma with
tumour necrosis of less than 5%. Immunohistochemical stain for IDH1
was negative. The resection margins were clear and Ki-67 proliferative
index was less than 1%. The patient completed 4 cycles of adjuvant
chemotherapy (Cisplatin & Doxorubicin). Bone transport continued
through-out his chemotherapy at 0–1 mm per day.
At 7 months after the index surgery, the nail reached its maximum
distraction distance of 80 mm. Approximately 35 mm of defect
remained. The second surgery was performed to revise the distal xation
of the nail. It was complicated by supercial wound infection which
resolved with debridement and antibiotics. Bone transport resumed 6
days after the second surgery and continued until docking was achieved
at 10 months (Fig. 5).
A third surgery was performed at 13 months where the Precice® nail
was exchanged with a static titanium nail. Corticocancellous bone graft
was taken from the iliac crest and xed to the docking site with a screw.
Cancellous bone graft from the iliac crest was placed at the regenerate.
Progressive weight bearing was allowed and at 26 months after the
index surgery, both the regenerate and docking sites showed bony
union.
The latest follow-up was at 34 months after index surgery. The range
of motion of his knee was 0–130◦, and 0–30◦for his ankle. He was able to
walk on hiking trails and could drive. The Musculoskeletal Tumour
Society Score [7] was 26/30. There was good consolidation of the cor-
ticotomy and docking sites (Fig. 6), and no evidence of recurrent or
metastatic disease on surveillance MRI and PET/CT scans.
3. Discussion
Reconstruction of distal tibia bone defects after resection of a ma-
lignant bone tumour is a challenge due to the difculty to obtain good
soft tissue coverage [8,9] and lack of established endoprosthesis for the
ankle joint; hence below-knee amputation was the standard treatment in
the past. Satisfactory functional results were reported [1,10] but
nowadays amputation is not easily accepted by the patient. Early
rehabilitation is offset by signicant psychological, social and prosthetic
issues in the longer term [10,11]. Many tumour centres can now perform
limb salvage surgery for most of their patients and a multitude of
reconstruction methods have been reported for distal tibia defects
without clear superiority [2–4].
The ideal reconstruction should have biological afnity, resistance to
infection, adequate biomechanical strength, durability and minimal
complications [12].
Fig. 3. Haematoxylin & Eosin stain of biopsy sample showing a chondroblastic
osteosarcoma [Original magnication 40×].
Fig. 4. Postoperative AP (A&B) & Lateral (C&D) radiographs showing resection of distal tibial tumour, and corticotomy, with Precice® nail and locking
plates inserted.
A.A. Eldesouqi et al.

International Journal of Surgery Case Reports 84 (2021) 106079
4
Endoprosthetic replacements restore skeletal integrity immediately
and allow early rehabilitation. However, late complications such as
infection, wear, and loosening subject the long-term survivor to revision
surgeries and the risk of delayed amputation [13–15].
Allografts can be used to reconstruct both small and large defects.
However, they may not be readily available [16] and obtaining a good
t is sometimes difcult. Moreover, their use is associated with high
rates of fracture (12–20%), nonunion (11–17%) and infection (12–15%)
[17,18].
Autografts provide a biological means of reconstruction for small
defects while large defects demand the use of vascularized bular graft
which is technically demanding and has signicant donor site
morbidity. In our case, the use of the ipsilateral bula as a pedicle graft
was not desirable as the resection margin included the medial cortex of
Fig. 5. AP (A) & lateral (B) radiographs at 10 months follow-up showing docking of the transported bone segment and regenerate formation.
A.A. Eldesouqi et al.

International Journal of Surgery Case Reports 84 (2021) 106079
5
the distal bula.
Bone recycling is an attractive option due to its simple technique and
exact size-matching. However, complete incorporation by living bone
takes a long time [19].
Distraction osteogenesis is a biological reconstructive technique that
can generate healthy bone [20–25]. When the regenerate ossies and
there is union at the docking site, skeletal integrity is restored with living
bone that has resistance against infection and can remodel to stress [26].
This technique has been widely used for the treatment of deformity,
limb-length discrepancy, osteomyelitis, non-union and traumatic bone
defects [3]. Traditional distraction osteogenesis requires the prolonged
use of external xator which carries a signicant risk of infection. This is
particularly undesirable for oncological patients who undergo chemo-
therapy. There is also some concern that chemotherapy may affect the
formation of regenerate. However, the evidence is discordant [27–31]
and no conclusion can be drawn at the present.
Tsuchiya et al. [29] reported the use of distraction osteogenesis for
reconstruction of bony defects after excision of tumour. In their series of
19 patients with defects in the femur or tibia, unilateral frame or Ilizarov
external xator was applied with additional intramedullary nail in
selected cases. Excellent results were achieved although complications
were present for 10 of the 19 patients.
With the development of a non-invasive growing intramedullary
nail, the risk of infection in distraction osteogenesis is reduced. Its use in
PABST was reported recently [32]. This is an attractive technique for the
musculoskeletal oncology patient as it provides skeletal stabilization
with all-internal xation and bone transport can be continued during
adjuvant chemotherapy by the patient at home. After the regenerate has
consolidated, this biological reconstruction should last the patient's
lifetime.
The reconstruction by this technique is limited by the distraction
length of the Precice® nail (80 mm). Hence a second operation was
required to reset the distracted nail. It also had to be exchanged to a
static titanium nail subsequently as it was not MRI-compatible. Union at
the docking site and consolidation of the regenerate are also variables
that may require close monitoring and additional bone grafting
procedure.
In our literature search for bone transport via an all-internal xation
technique, we found only a few examples of oncological patients and
only one involving the tibia [33]. We report this rst case of PABST after
distal tibia tumour resection to demonstrate the feasibility of an un-
common technique in this difcult area. The addition of a bridging-plate
gives adequate stability for a defect close to the joint. Given the risks
inherent to the prolonged use of external xator, many surgeons may not
consider bone transport as a viable option without awareness of this
technique, and may not attempt to salvage the native ankle joint. We
believe this is a useful tool to add to the armamentarium of the ortho-
paedic oncologist.
4. Conclusion
This is the rst case report of bone transport using the PABST tech-
nique after distal tibia tumour resection. It shows that this is a viable and
safe method of reconstruction for a difcult distal tibia large bone
defect. Despite the use of chemotherapy, regenerate was formed and
consolidated to give the patient excellent long-term prospects.
Sources of funding
This research did not receive any specic grant from funding
agencies in the public, commercial, or not-for-prot sectors.
Fig. 6. AP (A&B) & lateral (C&D) radiographs at 34 months follow-up showing consolidation of regenerate and union at docking site. Static titanium tibial nail has
been inserted.
A.A. Eldesouqi et al.

International Journal of Surgery Case Reports 84 (2021) 106079
6
Ethical approval
Ethical approval by the Institutional Review Board of the University
of Hong Kong/Hospital Authority Hong Kong West Cluster has been
obtained (Ref No. UW 15-414)
Consent
Written consent was obtained from the patient for publication of this
case report and accompanying images. A copy of the written consent is
available for review by the Editor-in-Chief of this journal on request.
CRediT authorship contribution statement
Ahmed A Eldesouqi: Drafting of manuscript.
Raymond Ching Hin Yau: Data collection, Revision of manuscript.
Wai-Yip Kenneth Ho: Data analysis and interpretation, Revision of
manuscript.
Ying-Lee Lam: Conception of work, Final approval.
Research Registration.
Not applicable.
Guarantor.
Raymond Ching Hin Yau, Ying-Lee Lam.
Declaration of competing interest
Nothing to declare.
Acknowledgements
We would like to acknowledge the help of Dr. Shek Wai Hung, Tony
(Department of Pathology, Queen Mary Hospital, Hong Kong) for his
help in preparation of the histology slide.
Provenance and peer review
Not commissioned, externally peer-reviewed.
References
[1] A.F. Mavrogenis, C.N. Abati, C. Romagnoli, P. Ruggieri, Similar survival but better
function for patients after limb salvage versus amputation for distal tibia
osteosarcoma, Clin. Orthop. Relat. Res. 470 (6) (2012) 1735–1748.
[2] R.J. Grimer, Surgical options for children with osteosarcoma, Lancet Oncol. 6 (2)
(2005) 85–92.
[3] K. Erler, C. Yildiz, B. Baykal, A.S. Atesalp, M.T. Ozdemir, M. Basbozkurt,
Reconstruction of defects following bone tumor resections by distraction
osteogenesis, Arch. Orthop. Trauma Surg. 125 (3) (2005) 177–183.
[4] P. Kunz, L. Bernd, Methods of biological reconstruction for bone sarcoma:
indications and limits, Recent Results Cancer Res. 179 (2009) 113–140.
[5] R.A. Agha, T. Franchi, C. Sohrabi, G. Mathew, A. Kerwan, The SCARE 2020
guideline: updating consensus surgical CAse REport (SCARE) guidelines, Int. J.
Surg. 84 (2020) 226–230.
[6] W.F. Enneking, S.S. Spanier, M.A. Goodman, A system for the surgical staging of
musculoskeletal sarcoma, Clin. Orthop. Relat. Res. (153) (1980) 106–120.
[7] W.F. Enneking, W. Dunham, M.C. Gebhardt, M. Malawar, D.J. Pritchard, A system
for the functional evaluation of reconstructive procedures after surgical treatment
of tumors of the musculoskeletal system, Clin. Orthop. Relat. Res. (286) (1993)
241–246.
[8] S. Shalaby, H. Shalaby, A. Bassiony, Limb salvage for osteosarcoma of the distal
tibia with resection arthrodesis, autogenous bular graft and Ilizarov external
xator, J. Bone Joint Surg. (Br.) 88 (12) (2006) 1642–1646.
[9] W. Ebeid, S. Amin, A. Abdelmegid, Y. Refaat, A. Ghoneimy, Reconstruction of
distal tibial defects following resection of malignant tumours by pedicled
vascularised bular grafts, Acta Orthop. Belg. 73 (3) (2007) 354–359.
[10] A.M. Davis, M. Devlin, A.M. Grifn, J.S. Wunder, R.S. Bell, Functional outcome in
amputation versus limb sparing of patients with lower extremity sarcoma: a
matched case-control study, Arch. Phys. Med. Rehabil. 80 (6) (1999) 615–618.
[11] M. Laitinen, J. Hardes, H. Ahrens, et al., Treatment of primary malignant bone
tumours of the distal tibia, Int. Orthop. 29 (4) (2005) 255–259.
[12] Y.Y. Abed, G. Beltrami, D.A. Campanacci, M. Innocenti, G. Scoccianti, R. Capanna,
Biological reconstruction after resection of bone tumours around the knee: long-
term follow-up, J. Bone Joint Surg. (Br.) 91 (10) (2009) 1366–1372.
[13] G. Ottaviani, R.S. Robert, W.W. Huh, N. Jaffe, Functional, psychosocial and
professional outcomes in long-term survivors of lower-extremity osteosarcomas:
amputation versus limb salvage, Cancer Treat. Res. 152 (2009) 421–436.
[14] L.M. Jeys, R.J. Grimer, S.R. Carter, R.M. Tillman, Risk of amputation following
limb salvage surgery with endoprosthetic replacement, in a consecutive series of
1261 patients, Int. Orthop. 27 (3) (2003) 160–163.
[15] D. Biau, F. Faure, S. Katsahian, C. Jeanrot, B. Tomeno, P. Anract, Survival of total
knee replacement with a megaprosthesis after bone tumor resection, J. Bone Joint
Surg. Am. 88 (6) (2006) 1285–1293.
[16] C. Delloye, O. Cornu, V. Druez, O. Barbier, Bone allografts: what they can offer and
what they cannot, J. Bone Joint Surg. Br. Vol. 89 (2007) 574–579.
[17] R.C. Thompson Jr., A. Garg, D.R. Clohisy, E.Y. Cheng, Fractures in large-segment
allografts, Clin. Orthop. Relat. Res. 370 (2000) 227–235.
[18] N. Dion, F.H. Sim, The use of allografts in musculoskeletal oncology, Instr. Course
Lect. 51 (2002) 499–506.
[19] K. Hayashi, H. Tsuchiya, N. Yamamoto, H. Minato, K. Tomita, Histological
examination of autoclaved bone removed 12 years after it was transplanted,
J. Orthop. Sci. 10 (4) (2005) 425–429.
[20] H. Tsuchiya, M.E. Abdel-Wanis, K. Tomita, Biological reconstruction after excision
of juxta-articular osteosarcoma around the knee: a new classication system,
Anticancer Res. 26 (1B) (2006) 447–453.
[21] H. Tsuchiya, K. Tomita, Distraction osteogenesis for treatment of bone loss in the
lower extremity, J. Orthop. Sci. 8 (1) (2003) 116–124.
[22] H. Tsuchiya, M. Abdel-Wanis, S. Kitano, K. Sakurakichi, T. Yamashiro, K. Tomita,
The natural limb is best: joint preservation and reconstruction by distraction
osteogenesis for high-grade juxta-articular osteosarcomas, Anticancer Res. 22 (4)
(2002) 2373–2376.
[23] H. Tsuchiya, M.E. Abdel-Wanis, K. Sakurakichi, T. Yamashiro, K. Tomita,
Osteosarcoma around the knee. Intraepiphyseal excision and biological
reconstruction with distraction osteogenesis, J. Bone Joint Surg. (Br.) 84 (8) (2002)
1162–1166.
[24] K. Hayashi, H. Tsuchiya, N. Yamamoto, A. Takeuchi, K. Tomita, Functional
outcome in patients with osteosarcoma around the knee joint treated by minimised
surgery, Int. Orthop. 32 (1) (2008) 63–68.
[25] H. Tsuchiya, A.F. Morsy, H. Matsubara, K. Watanabe, M.E. Abdel-Wanis, K. Tomita,
Treatment of benign bone tumours using external xation, J. Bone Joint Surg. (Br.)
89 (8) (2007) 1077–1083.
[26] K. Watanabe, H. Tsuchiya, N. Yamamoto, et al., Over 10-year follow-up of
functional outcome in patients with bone tumors reconstructed using distraction
osteogenesis, J. Orthop. Sci. 18 (1) (2013) 101–109.
[27] M. Subasi, A. Kapukaya, C. Kesemenli, T.A. Balci, H. Buyukbayram, M. Ozates,
Effect of chemotherapeutic agents on distraction osteogenesis. An experimental
investigation in rabbits, Arch. Orthop. Trauma Surg. 121 (7) (2001) 417–421.
[28] J. Ca˜
nadell, M. San-Julian, J. Cara, F. Forriol, External xation in tumour
pathology, Int. Orthop. 22 (2) (1998) 126–130.
[29] H. Tsuchiya, K. Tomita, K. Minematsu, Y. Mori, N. Asada, S. Kitano, Limb salvage
using distraction osteogenesis. A classication of the technique, J. Bone Joint Surg.
(Br.) 79 (3) (1997) 403–411.
[30] B. Demiralp, T. Ege, O. Kose, Y. Yurttas, M. Basbozkurt, Reconstruction of
intercalary bone defects following bone tumor resection with segmental bone
transport using an Ilizarov circular external xator, J. Orthop. Sci. 19 (6) (2014)
1004–1011.
[31] H. Tsuchiya, T. Shirai, A.F. Morsy, et al., Safety of external xation during
postoperative chemotherapy, J. Bone Joint Surg. (Br.) 90 (7) (2008) 924–928.
[32] G. Barinaga, A.M. Beason, M.P. Gardner, Novel surgical approach to segmental
bone transport using a magnetic intramedullary limb lengthening system, J. Am.
Acad. Orthop. Surg. 26 (22) (2018) e477–e482.
[33] U.K. Olesen, T. Nygaard, D.E. Prince, et al., Plate-assisted bone segment transport
with motorized lengthening nails and locking plates: a technique to treat femoral
and tibial bone defects, J. Am. Acad. Orthop. Surg. Glob. Res. Rev. 3 (8) (2019),
e064.
A.A. Eldesouqi et al.