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: afm@otenet.gr; andreasfmavrogenis@yahoo.gr

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

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Eur J Orthop Surg Traumatol (2012) 22 (Suppl 1):S199–S204

DOI 10.1007/s00590-012-1013-0

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

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123

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

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

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123

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.

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