![Page 1: Successful treatment of aneurysmal bone cyst of the proximal fibula with embolization](https://reader031.vdocuments.us/reader031/viewer/2022020300/575094d41a28abbf6bbc7fd4/html5/thumbnails/1.jpg)
UP-TO DATE REVIEW AND CASE REPORT
Successful treatment of aneurysmal bone cyst of the proximalfibula with embolization
Andreas F. Mavrogenis • Giuseppe Rossi •
Eugenio Rimondi • Pietro Ruggieri
Received: 12 April 2012 / Accepted: 2 May 2012 / Published online: 25 May 2012
� Springer-Verlag 2012
Abstract We present an 8-year-old girl with evolution of
an aneurysmal bone cyst of the proximal fibula after int-
ralesional surgery, treated successfully with one selective
transfemoral embolization with N-2-butyl-cyanoacrylate.
By the first 5 days, the patient experienced complete pain
relief. Post-embolization imaging follow-up showed pro-
gressive homogenous trabecular bone formation and
gradual reduction of the size of the lesion. These findings
canceled our initial consideration for surgical treatment.
Three years after the embolization, healing of the lesion
and remodeling of the proximal fibula were observed
without evidence of recurrence. Our successful results with
excellent response seen 3 years after embolization are in
favor and should increase the awareness of surgeons
regarding embolization for aneurysmal bone cysts in dif-
ficult anatomic locations such as the proximal fibula.
Keywords Aneurysmal bone cyst � Proximal fibula �Selective arterial embolization
Introduction
Aneurysmal bone cysts are expansile, hemorrhagic bone
lesions of unknown origin and more than 50 % occur in
the long bones [1]. The fibula is a rare location; the
proximal part even more rare [2–4]. Given the benign
biological behavior of aneurysmal bone cysts, intrale-
sional procedures have been the most commonly used
treatment options [3, 4]. Resection is primarily used in
expendable bones and patients with active or aggressive
and recurrent tumors [3]. However, surgical treatment has
been associated with complications [3, 5] and recurrences
ranging from 10 to 59 % [2, 6, 7]. Recurrence is usually
seen within 24 months following the original treatment;
young age of presentation [1, 3] and intralesional surgery
[1, 2] have been associated with a higher risk of local
recurrence [9]. In certain locations such as the proximal
fibula, higher rates of surgical complications including
lateral knee instability and neurovascular injury have been
reported [3].
Recently, minimally invasive treatments such as
cryotherapy, radiation therapy, percutaneous intralesional
injection of calcitonin, methylprednisolone or sclerosing
agents [5, 8, 9], and selective arterial embolization [2, 8,
11, 12] have been reported for aneurysmal bone cysts.
Selective embolization of aneurysmal bone cysts of the
proximal fibula has not been previously reported. This
article presents an 8-year-old girl with evolution of
an aneurysmal bone cyst of the proximal fibula after
intralesional surgery, treated successfully with one
selective arterial embolization. The parents gave written
informed consent for patient’s data to be included in
this study. This study was approved by the Institu-
tional Review Board/Ethics Committee of the authors’
institution.
A. F. Mavrogenis (&)
First Department of Orthopaedics, Athens University Medical
School, 41 Ventouri Street, 15562 Holargos, Athens, Greece
e-mail: [email protected]; [email protected]
G. Rossi � E. Rimondi
Department of Interventional Angiographic Radiology,
Istituto Ortopedico Rizzoli, Via Di Barbiano 1/10,
40136 Bologna, Italy
P. Ruggieri
Department of Orthopaedics, Istituto Ortopedico Rizzoli,
Bologna, Italy
123
Eur J Orthop Surg Traumatol (2012) 22 (Suppl 1):S199–S204
DOI 10.1007/s00590-012-1013-0
![Page 2: Successful treatment of aneurysmal bone cyst of the proximal fibula with embolization](https://reader031.vdocuments.us/reader031/viewer/2022020300/575094d41a28abbf6bbc7fd4/html5/thumbnails/2.jpg)
Case report
An 8-year-old girl presented to the authors’ institution with
a painful palpable mass at the left proximal fibula. She had
imaging and histological diagnosis of aneurysmal bone
cyst, and underwent intralesional surgery (Fig. 1). Six
months later, imaging of the left knee showed an evolving,
expansile osteolytic lesion of the proximal fibula with
fluid–fluid levels (Fig. 2). Biopsy of the lesion was repe-
ated at this time and confirmed the previous diagnosis.
Because of the location and size of the lesion, and the age
of the patient, selective arterial embolization was decided
aiming to gain control of the destructive process before a
possible reconstructive procedure.
Under general anesthesia, digital subtraction angiogra-
phy was performed through the contralateral common
femoral artery to obtain a vascular map of the patholog-
ical vessels (Fig. 3a). Two feeding arteries were identi-
fied, one originating from a collateral branch of the
anterior tibial artery (Fig. 3b) and the second from a
segmental branch of tibioperoneal trunk (Fig. 3c). The
feeding vessels were selectively catheterized with a 4
French diagnostic catheter and a microcatheter (Cordis
Corporation, Johnson & Johnson, East Bridgewater, NJ,
USA). Embolization was performed using as embolizing
agent N-2-butyl-cyanoacrylate (NBCA, Glubran 2, GEM,
Viareggio, Italy) in 33 % lipiodol ultrafluid, ‘‘sand-
wiched’’ with 5 % glucosate solution to prevent poly-
merization with blood. One flacon (1 ml) of NBCA was
mixed with 2 ml of 33 % lipiodol. From the mixture,
1 ml was aspirated in an insulin (1 ml) syringe, and
0.1–0.2 ml of the aspirate mixture was injected ‘‘sand-
wiched’’ with 2 ? 2 ml of 5 % glucosate solution under
fluoroscopic control. Then, angiography was performed to
evaluate occlusion and remaining pathological vasculature
(Fig. 3d).
Post-embolization recovery of the patient was unevent-
ful; immediately postoperatively she had increased pain
that was treated with oral analgesics. The patient was
discharged from hospital the next day with significant
Fig. 1 a Anteroposterior radiograph of the left knee shows an
expansile osteolytic lesion involving the proximal fibula. b Sagittal
T1-weighted magnetic resonance (MR) imaging shows an
intermediate signal intensity lesion with fluid–fluid levels. Biopsy
showed aneurysmal bone cyst, and surgical treatment was performed
S200 Eur J Orthop Surg Traumatol (2012) 22 (Suppl 1):S199–S204
123
![Page 3: Successful treatment of aneurysmal bone cyst of the proximal fibula with embolization](https://reader031.vdocuments.us/reader031/viewer/2022020300/575094d41a28abbf6bbc7fd4/html5/thumbnails/3.jpg)
Fig. 2 Six months after surgical treatment, a anteroposterior and b lateral radiographs of the left knee and c sagittal T1-weighted MR imaging
show evolution of the aneurysmal bone cyst
Fig. 3 a Digital substraction angiography through selective cathe-
terization using a Seldinger catheter inserted by the right common
femoral artery revealed two feeding vessels (arrows 1 and 2).
b Selective catheterization of the first feeding vessel (1) originating
from a collateral branch of the anterior tibial artery. c Selective
catheterization of the second feeding vessel (2) originating from a
segmental branch of tibioperoneal trunk. d Post-embolization angi-
ography shows occlusion of the feeding arteries
Eur J Orthop Surg Traumatol (2012) 22:S199–S204 S201
123
![Page 4: Successful treatment of aneurysmal bone cyst of the proximal fibula with embolization](https://reader031.vdocuments.us/reader031/viewer/2022020300/575094d41a28abbf6bbc7fd4/html5/thumbnails/4.jpg)
improvement of pain. By the first 5 days, the patient
experienced complete pain relief. Post-embolization
imaging follow-up showed progressive homogenous tra-
becular bone formation and gradual reduction of the size of
the lesion. These findings canceled our initial consideration
for surgical treatment. At 3 years, the patient was symp-
tom-free; radiograph of the knee showed reduction of size
and ossification of the lesion, and remodeling of the
proximal fibula without evidence of recurrence (Fig. 4).
Discussion
Aneurysmal bone cysts of the proximal fibula are rare;
embolization of aneurysmal bone cysts of the proximal
fibula has not been previously reported [2–4]. In this brief
report, we presented a patient with evolution of an aneu-
rysmal bone cyst after intralesional surgery, subsequently
treated successfully with one selective embolization. Our
successful results with excellent response seen 3 years after
embolization are in favor and should increase the aware-
ness of surgeons regarding embolization for such lesions in
difficult anatomic locations such as the proximal fibula.
Traditionally, intralesional surgery with or without adju-
vants has been the most common treatment for aneurysmal
bone cysts [2, 3, 5, 8, 9]. However, persistence and
evolution, or recurrence of the cysts is not uncommon after
intralesional surgery [1, 2], as in the present patient.
Probably, the clinical behavior of aneurysmal bone cysts is
more aggressive in younger patients, with higher recur-
rence rates after surgical treatment [3, 6]. Segmental, en
bloc resection of the involved bone has been suggested
for active, aggressive, and recurrent lesions [2]. In the
proximal fibula, intraarticular ‘‘en bloc’’ resection includes
removal of the proximal part of the fibula with detachment
of the biceps femoris tendon, the fibular collateral liga-
ment, the anterior tibial and peronei muscles [14–17], and
disruption of the attachment of the biceps femoris muscle
to the iliotibial band [16, 17]. These can lead to lateral
instability of the knee [16], and weakness in inversion and
eversion of the foot [18]. Motion of the knee joint is usu-
ally spared [18, 19]. However, accurate surgical recon-
struction of the biceps femoris tendon and fibular collateral
ligament to the lateral aspect of the tibia is necessary.
Additionally, the patients should follow a rehabilitation
program as after an acute injury of the lateral structures of
the knee. Other potential complications related to surgical
treatment include skeletal deformities from growth plate
injury [5], infection, vascular and peroneal nerve injury
related to the surgical exposure and the tumor expansion,
Fig. 4 At the latest evaluation,
3 years after embolization,
a anteroposterior and b lateral
radiographs of the left knee
show further reduction of size
and ossification of the lesion,
and remodeling of the proximal
fibula without evidence of local
recurrence
S202 Eur J Orthop Surg Traumatol (2012) 22 (Suppl 1):S199–S204
123
![Page 5: Successful treatment of aneurysmal bone cyst of the proximal fibula with embolization](https://reader031.vdocuments.us/reader031/viewer/2022020300/575094d41a28abbf6bbc7fd4/html5/thumbnails/5.jpg)
and pulmonary embolism [3]. It is also possible that the
peroneal nerve is tethered by fascial bands to the proximal
fibula, and may be further compromised by surgical
resection.
The main advantage of selective arterial embolization is
the avoidance of surgery, hence avoidance of complex
reconstructive procedures and postoperative complications
[12]. A success rate of up to 94 % [13] and a recurrence
rate of up to 39 % have been reported with embolization
[10, 13]. Persistence or recurrence after embolization has
been associated with size greater than 5 cm of the aneu-
rysmal bone cysts [13]; in these cases, repeat embolization
can be performed successfully [13]. The overall risk of
embolization-related complications ranges up to 6 %.
These complications include dissection of the femoral
artery at the site of transarterial catheterization, pain due to
ischemic tumor necrosis, accidental embolization into non-
tumor vessels, infection, and post-embolization syndrome
[10, 13]. The post-embolization syndrome has been
reported in 18 to 86 % of cases [10]. Embolization of
adjacent normal vessels can result in a large zone of tissue
loss and may be associated with risk of nerve palsy, skin
breakdown, and subcutaneous or muscle necrosis; tissue
ischemia may lead to infection. To avoid embolizing
adjacent normal tissue, careful evaluation in diagnostic
angiography of the relationship of the feeding arteries with
the adjacent vessels, selective catheterization, and embo-
lization of the pathological feeding arteries to the lesion
with the most appropriate embolic agent is necessary [10].
The risk of complications is higher in certain anatomic
regions such as (1) the femur to avoid embolizing the lat-
eral circumflex femoral artery and the supply to the sciatic
nerve and the lateral cutaneous nerve of the thigh, (2) the
humerus to avoid the posterior circumflex humeral artery,
(3) the spine to avoid the Adamkiewicz artery that origi-
nates between the T5 and L2 vertebra, and (4) the pelvis to
avoid the posterior branch of the internal iliac artery and
the inferior gluteal artery [10, 13].
Complications related to the embolic agent may also
occur. Embolic agents include gelfoam, polyvinyl alcohol
(PVA) particles, liquid (absolute alcohol), coils, tissue
adhesives, ethanol, and microfibrillar collagen [10]. Major
considerations for choosing an embolic agent are speed and
reliability of delivery, duration of occlusive effect, preser-
vation of normal tissue, and operator’s experience [10].
Liquid embolic agents offer the advantages of low viscosity
for easy injection through small catheters or catheters with
many bends through tortuous vessels. NBCA or ‘‘liquid
glue’’ is a liquid embolic agent that spreads according to its
polymerization time and the vascular flow, and can pass
through bent catheters navigating tortuous vessels. Addi-
tionally, NBCA in lipiodol is densely radiopaque that allows
for its exact site of occlusion to be observed [10, 13].
In the present patient, because of the young age, the
large lesion that showed evolution after intralesional sur-
gery and the difficult anatomic location, embolization was
decided as preoperative adjuvant to decrease the vascu-
larity of the cyst and facilitate surgical treatment before a
reconstructive procedure. We used NBCA for controlled
embolization and permanent occlusion of the entire path-
ological vasculature, and complete devascularization of the
lesion. Bolus administration of small doses (0.1–0.2 ml) of
NBCA in 33 % lipiodol sandwiched with 5 % glucosate
solution under fluoroscopic control, followed by arteriog-
raphy provided for the efficacy and safety of the procedure;
post-embolization complications were not observed. Dur-
ing imaging follow-up, the lesion stopped to expand; pro-
gressive reduction of size and ossification altered our
decision for surgery. Three years after one embolization,
further reduction of size and ossification of the lesion was
observed, without evidence of recurrence. Our successful
results show the potential value of embolization and should
increase the awareness of surgeons regarding embolization
for aneurysmal bone cysts in difficult anatomic locations
such as the proximal fibula.
Conflict of interest No funds were received in support of this study.
References
1. Zehetgruber H, Bittner B, Gruber D et al (2005) Prevalence of
aneurysmal and solitary bone cysts in young patients. Clin Orthop
Relat Res 439:136–143
2. Vergel De Dios AM, Bond JR, Shives TC et al (1992) Aneu-
rysmal bone cyst. A clinicopathologic study of 238 cases. Cancer
69(12):2921–2931
3. Basarir K, Piskin A, Guclu B et al (2007) Aneurysmal bone cyst
recurrence in children: a review of 56 patients. J Pediatr Orthop
27(8):938–943
4. Karkuzhali P, Bhattacharyya M, Sumitha P (2007) Multiple soft
tissue aneurysmal cysts: an occurrence after resection of primary
aneurysmal bone cyst of fibula. Indian J Orthop 41(3):246–249
5. Mankin HJ, Hornicek FJ, Ortiz-Cruz E et al (2005) Aneurysmal
bone cyst: a review of 150 patients. J Clin Oncol 23(27):6756–
6762
6. Freiberg AA, Loder RT, Heidelberger KP, Hensinger RN (1994)
Aneurysmal bone cysts in young children. J Pediatr Orthop
14(1):86–91
7. Bollini G, Jouve JL, Cottalorda J et al (1998) Aneurysmal bone
cyst in children: analysis of twenty-seven patients. J Pediatr
Orthop B 7(4):274–285
8. Cottalorda J, Kohler R, Chotel F et al (2005) Recurrence of
aneurysmal bone cysts in young children: a multicenter study.
J Pediatr Orthop B 14(3):212–218
9. Cottalorda J, Bourelle S (2007) Modern concepts of primary
aneurysmal bone cyst. Arch Orthop Trauma Surg 127(2):105–114
10. Green JA, Bellemore MC, Marsden FW (1997) Embolization in
the treatment of aneurysmal bone cysts. J Pediatr Orthop
17(4):440–443
11. Dormans JP, Hanna BG, Johnston DR, Khurana JS (2004) Sur-
gical treatment and recurrence rate of aneurysmal bone cysts in
children. Clin Orthop Relat Res 421:205–211
Eur J Orthop Surg Traumatol (2012) 22:S199–S204 S203
123
![Page 6: Successful treatment of aneurysmal bone cyst of the proximal fibula with embolization](https://reader031.vdocuments.us/reader031/viewer/2022020300/575094d41a28abbf6bbc7fd4/html5/thumbnails/6.jpg)
12. Marushima A, Matsumaru Y, Suzuki K et al (2009) Selective
arterial embolization with n-butyl cyanoacrylate in the treatment
of aneurysmal bone cyst of the thoracic vertebra: a case report.
Spine 34(6): E230–E234
13. Rossi G, Rimondi E, Bartalena T et al (2010) Selective arterial
embolization of 36 aneurysmal bone cysts of the skeleton with
N-2-butyl cyanoacrylate. Skeletal Radiol 39(2):161–167
14. Grood ES, Noyes FR, Butler DL, Suntay WJ (1981) Ligamentous
and capsular restraints preventing straight medial and lateral
laxity in intact human cadaver knees. J Bone Joint Surg Am
63A:1257–1269
15. Seering WP, Piziali RL, Nagel DA, Schurman DJ (1980) The
function of the primary ligaments of the knee in varus-valgus and
axial rotation. J Biomech 13:785–794
16. Malawer MM (1984) Surgical management of aggressive and
malignant tumors of the proximal fibula. Clin Orthop Relat Res
186:172–181
17. Draganish LF, Nicholas RW, Shuster JK et al (1991) The effects
of resection of the proximal fibula on stability of the knee and on
gait. J Bone Joint Surg Am 73A:575–583
18. Youdas JW, Wood MB, Cahalan TD, Chao EYS (1988) A
quantitative analysis of donor site morbidity after vascularized
fibula transfer. J Orthop Res 6:621–629
19. Scranton PE, McMaster JH, Kelly E (1976) Dynamic fibular
function. Clin Orthop Relat Res 118:76–81
S204 Eur J Orthop Surg Traumatol (2012) 22 (Suppl 1):S199–S204
123