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Case Report
Vascularized fibular graft and anconeus interposition arthroplasty in a case
of recurrent aggressive aneurysmal bone cyst of proximal radius: A
case report
Anil Regmi
a
, Sunit Vardhan
a
, Akshay Kapoor
b
, Bishwa Bandhu Niraula
a
, Shivam Bansal
a
,
Mohit Dhingra
a
,
*
a
Department of Orthopedics, AIIMS, Rishikesh, Uttarakhand, 249203, India
b
Department of Burn and Plastic Surgery, AIIMS, Rishikesh, Uttarakhand, 249203, India
ARTICLE INFO
Keywords:
Aneurysmal bone cyst
Vascularized fibular graft
Anconeus interposition arthroplasty
Limb salvage surgery
Reconstructive surgery
Case report
ABSTRACT
Introduction: Large aggressive tumors need wide resection and reconstructions. The choice of reconstruction of the
defects is limited in pediatric population. Vascular fibula offers a promising graft in these cases. Biological
interpositional arthroplasty with the use of local muscle (anconeus) is a promising approach to achieve acceptable
range of movement of elbow and forearm.
Case presentation: A ten-year-old male child with recurrent ABC of the left proximal radius had undergone en bloc
tumor resection, and reconstruction by vascularized fibular grafting and proximal radio-ulnar joint reconstructed
with anconeus interposition arthroplasty, showed good clinical results and excellent radiographic results ac-
cording to the Tang system at 6 month follow-up.
Discussion and conclusion: Reconstruction of the resected long bone segment by vascularized fibular graft and
Anconeus interposition arthroplasty to reconstruct the radial head in a fibular graft is a convenient alternative to
prostheses.
Level of evidence: V.
1. Introduction
Aneurysmal Bone Cyst (ABC)s are considered primary lesions in
approximately 70% of cases, with most cases occurring at the end of long
bones with male predominance.
1
Patient usually presents in early
twenties in male predominance with the complains of pain, soft-tissue
swelling, or a palpable expansile mass.
2
They present as a radiolucent
cystic lesion in metaphyseal boundaries of the long bone on a plain
radiograph.
3
In addition to plain radiograph, contrast-enhanced MRI
shows areas of cystic lesions with areas of solid blood component that
indicate fluid-fluid interface.
4,5
Core biopsy and histopathological ex-
amination guide to the final diagnosis.
6
Traditionally ABC was treated surgically where lesions in bones such
as fibula, clavicle, and distal ulna were excised and lesions in other long
bones were curetted and bone grafted.
7
Presently there has been a sig-
nificant change in the approach from aggressive surgical approach to
sclerotherapy and curopsy.
8
But these modalities are being used for small
to large lesions and areas which are not suitable for aggressive resection.
9
In general, intralesional curettage and bone grafting are used as man-
agement, both with and without adjuvant therapy.
10
As part of adjuvant
therapy, microscopic disease contamination within the tumor bed is
treated to reduce the chances of local recurrence.
11
Wide resection and
reconstruction can be considered for lesions that have destroyed the
metaphyseal bone in periarticular areas.
12
Reconstruction plays an important role in musculoskeletal oncology
where limb salvage surgery is targeted. Biological methods such as bone
grafts and distraction osteogenesis are preferred used for reconstruc-
tion.
12
The use of this method become more important in pediatric age
group. Use of vascularized bone grafts are preferred than conventional
grafts in the presence of large defects.
13
In the presence of involvement of
near joint and young age arthroplasty with the help of available bio-
logical options is used to regain movements.
14
This report presents a case of a histologically proven massive recur-
rent aneurysmal bone cyst of the left proximal radius where wide en bloc
* Corresponding author.
E-mail addresses: regmiaanil@gmail.com (A. Regmi), sonuvardhan09@gmail.com (S. Vardhan), akshay.psurg@aiimsrishikesh.edu.in (A. Kapoor),
bishwa8bangladesh@gmail.com (B.B. Niraula), shivam.ban19@gmail.com (S. Bansal), mohit.orth@aiimsrishikesh.edu.in,modisbanu77@gmail.com (M. Dhingra).
Contents lists available at ScienceDirect
Journal of Orthopaedic Reports
journal homepage: www.journals.elsevier.com/journal-of-orthopaedic-reports
https://doi.org/10.1016/j.jorep.2023.100143
Received 28 January 2023; Accepted 17 February 2023
Available online 22 February 2023
2773-157X/©2023 The Author(s). Published by Elsevier B.V. on behalf of Prof. PK Surendran Memorial Education Foundation. This is an open access article under the
CC BY license (http://creativecommons.org/licenses/by/4.0/).
Journal of Orthopaedic Reports 2 (2023) 100143
resection was done and defect created was grafted with autologous
vascularized fibula. The proximal radio-ulnar joint was recreated by
interposition arthroplasty of anconeus between ulna and proximal aspect
of grafted fibula.
2. Case report
A 10-year-old male child with right dominance, presented with
swelling in left proximal forearm for 1.5 years. He gave a history of
curettage for the same lesion one year back at an elsewhecentertre. No
history of significant trauma, no significant family history was present.
3. Clinical findings
At presentation there was a globular swelling in the proximal third of
the left forearm of size 10x8 cm, which extended 2 cm below the cubital
fossa up to 8 cm proximal to the wrist joint. A primarily healed surgical
scar of 8x2 cm was visible over the swelling on the volar aspect of the left
proximal forearm. Venous prominence or any visible pulsations were
absent over the swelling (Fig. 1).
4. Diagnostic assessment
Radiographs revealed an expansile lytic lesion with bony septa over
the proximal end of the radius (Fig. 2). Magnetic Resonance Imaging was
performed as pre-operative planning, which revealed T1 weighted
hypointense, well-defined, multiloculated, expansile, lytic lesion, and
hyperintense cystic lesion in T2. The lesion was of the average size of
8.5x5x5.5cm, extended from the proximal radius remaining the unaf-
fected radial epiphysis of 3mm. (Fig. 3). The diagnosis was again
confirmed with histopathology.
5. Therapeutic intervention
With due informed consent from patient and his parents wide en-bloc
resection of the tumor and reconstruction with vascularized fibula and
interposition arthroplasty was performed. The tumor was approached
incorporating the previous surgical scar. All the vital soft tissue structures
were identified, tagged, and separated from the tumor tissue (Fig. 4).
Then, tumor tissue with a margin of 2 cm, including the radial head and
was resected. The total length of resected segment was 12 cm. (Fig. 5).
The reconstruction was done by vascularized fibular graft from the
Fig. 1. Clinical Image of left proximal forearm showing globular swelling in
proximal third.
Fig. 2. Plain Radiographs of left proximal forearm showing an Expansile lytic
lesion with bony septa over the proximal end of the radius.
Fig. 3. Pre-operative MRI of left elbow with forearm showing a lesion; A: T1 weighted hypointense, well-defined, multiloculated, expansile, lytic lesion; B: hyper-
intense cystic lesion in T2; C: unaffected radial epiphysis of 3mm.
A. Regmi et al. Journal of Orthopaedic Reports 2 (2023) 100143
2
contralateral side and anastomosed with the radial artery (figure-6).
Dynamic compression plate was used for fixation of fibula with the left
radius. Anconeus was mobilized and wrapped around the proximal fibula
and sutured over itself (figure-7).
6. Follow-up and outcomes
The postoperative period was uneventful, except for weakness of
thumb and index finger extension. Grafted tissue remained viable. Limb
was maintained in slab for six weeks with elbow in maximum supination.
Post operative radiographs of immediate post operative, and six months
were satisfactory (figure-8).
Radiological and functional assessment of the graft was done by Tang
system.
12
At final follow up, good clinical result and excellent radio-
graphic results according to Tang system. The functional assessment of
elbow flexion-extension and forearm supination and protonation move-
ment was measured. Elbow flexion-extension of 0–110, with forearm
supination-neutral-pronation movement of 80-0-70was found
(figure-9), and the patient was able to perform routine daily activities.
The Mayo Elbow performance score was 90, MSTS scoring of the upper
limb was 18 at three months and 23 at six months. And on six-month of
Fig. 4. Intra-Operative image showing tagged, and separated vital soft tissue
structures from the tumor tissue.
Fig. 5. Intra-operative image showing 12 cm of resected tumor including the radial head.
Fig. 6. Intra-operative image showing: A, Harvesting fibular graft; B, harvested vascularized fibula; C, Bony fixation of distal radius with fibula by dynamic
compression plate; D, anastomosis of vascularized fibular graft with the radial artery.
A. Regmi et al. Journal of Orthopaedic Reports 2 (2023) 100143
3
follow-up, observed no complications, Grade 1 according to Clavien
Dindo classification.
15
7. Discussion
ABCs are considered primary lesions in approximately 70% of cases,
most commonly occurs at the femur, tibia, humerus, and fibula.
1
The
patients presents with primary symptoms of pain, soft-tissue swelling, or
a palpable expansile mass.
3
In our case, 10 years old male child presented
with pain and soft tissue swelling which progressed to palpable mass at
unusual site of proximal forearm, involving proximal radius.
Percutaneous sclerotherapy with polidocanol is a safe alternative to
conventional surgery for the treatment of ABC in outpatient basis.
16
Rastogi S. et al.,
17
treated 72 patient on ABC by percutaneous sclero-
therapy with polidocanol with average injection of 3
1–5
with successful
radiological healing in 76.6% and mean clinical response of 84.5% and
recurrence rate of 2.8%. In our case, the lesion was large and had a
recurrence so wide enbloc tumor resection was done.
Vascularized fibular grafts are considered to have promising results
for biological reconstruction of massive bony defects.
18
Minami A et al.
13
performed 104 vascularized fibular grafts in 102 patients where primary
bony union occurred in 84%, with mean time to union of 15.5 weeks. In
our case, Vascularized fibular graft was performed for the reconstruction
of proximal radius after the excision of tumor, where bony union
occurred at the follow up of 6 month with good clinical result and
excellent radiographic results according to Tang system.
12
Baghdadi YM et al.
19
suggested that the anconeus arthroplasty to
address pathology at the radiocapitellar and/or proximal radioulnar
joint, as an alternatives of radial head replacement for marked proximal
radius bone loss. In our study, the radial head was excised and the
proximal end of vascularized fibular bone segment was wrapped with
anconeus to provide smooth surface for flexion/extension and supina-
tion/protonation movement. It also helps to avoid formation of any bony
bridge between opposing surface of bones and hinder movement.
14
On a thorough search of literature, there are only few literatures
available on anconeus interposition arthroplasty. In 2014, Baghdadi et al.
performed a retrospective study on 29 patients with an anconeus inter-
position arthroplasty, where mayo elbow performance score was signif-
icantly increased in the postoperative phase and had an excellent or good
outcome.
19
Similarly, in 2018, Rahmi et al. performed a retrospective
study on 23 patients with an anconeus interposition arthroplasty, where
the range of motion was significantly improved from preoperatively to
postoperatively.
20
Our case is unique in.
The rarity of ABC in the proximal radius
Use of vascularized fibular graft for reconstruction of resected prox-
imal radius
Fig. 7. Intra-operative image showing reconstruction of radial head on proximal end of grafted vascularized fibular graft by Anconeus interposition arthroplasty.
Fig. 8. Post-operative radiograph, showing A: Immediate post-operative radiograph showing reconstructed proximal radius with fibula with its adequate fixation with
distal end of radius, B: 6 month follow up radiograph of patient showing radiological bony union of the distal segment of the radius with a vascularized fibular graft.
A. Regmi et al. Journal of Orthopaedic Reports 2 (2023) 100143
4
Anconeus interposition arthroplasty to prevent synostosis between
fibula and ulna and also between fibula and humerus, thus providing
a pseudoarthrosis kind of joint.
To conclude, the limb salvage approach involving a multidisciplinary
team is the current recommended treatment for any bone tumors. En bloc
resection of tumor is recommended in view of large size and recurrence.
Biological reconstruction with vascularized fibula and interposition
arthroplasty is a promising approach to manage these tumors in imma-
ture skeletons.
Financial support and sponsorship
None.
Informed consent
Informed consent was obtained from the patient for publication of
this case report. On request, a copy of the written consent is available for
review by the Editor-in-Chief of this journal.
Ethical clearance
Intuitional ethical committee approved the study (retrospective, case
report) for paper publication. A copy of the ethical clearance is available
for review by the Editor-in-Chief of this journal on request.
Authors contribution
A. R. - Planning of study, writing, and revising the manuscript. S. V. -
Planning of study, writing the manuscript. A. K. –Data collection, Writing
the manuscript. B. B. N –Data Management. V. M. - Data Management.
M. D - Planning of study, writing, and revising the manuscript.
Declaration of competing interest
There is no conflict of interest of any kind with the research or its
outcome among the investigators.
Acknowledgment
None.
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