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1212 THE JOURNAL OF BONE AND JOINT SURGERY

Treatment of aneurysmal bone cysts withpercutaneous sclerotherapy usingpolidocanol

A REVIEW OF 72 CASES WITH LONG-TERM FOLLOW-UP

S. Rastogi,M. K. Varshney,V. Trikha,S. A. Khan,B. Choudhury,R. Safaya

From the All India

Institute of Medical

Sciences, New Delhi,India

S. Rastogi, MS, DNB, PMR,

Orthopaedic Surgeon,

Professor

M. K. Varshney, MS, DNB,

Senior Resident

V. Trikha, MS, Assistant

Professor

S. A. Khan, MS, DNB, MCh,

Assistant Professor

B. Choudhury, MS, Assistant

Professor

Department of Orthopaedics

R. Safaya, MD, Professor

Department of Pathology

C11/13 Ansari Nagar, All India

Institute of Medical Sciences,New Delhi 110029, India.

Correspondence should be sent

to Professor S. Rastogi; e-mail:

[email protected]

©2006 British Editorial Society

of Bone and Joint Surgery

doi:10.1302/0301-620X.88B9.

17829 $2.00

J Bone Joint Surg [Br]

2006;88-B:1212-16.

Received 28 February 2006;

Accepted after revision 12 April

2006

Aneurysmal bone cyst is a rare tumour-like lesion which develops during growth. Our aim

was to determine the efficacy of the administration of percutaneous intralesional 3%

polidocanol (hydroxypolyaethoxydodecan) as sclerotherapy.

Between July 1997 and December 2004 we treated 72 patients (46 males, 26 females)

with a histologically-proven diagnosis of aneurysmal bone cyst, at various skeletal sites

using this method. The sclerotherapy was performed under fluoroscopic guidance and

general anaesthesia or sedation and local anaesthesia. The mean follow-up period was 34

months (26.5 to 80). The patients were evaluated using the Enneking system for functional

assessment and all the lesions were radiologically quantified into four grades.

The mean age of patients was 15.6 years (3 to 38) and the mean number of injectionswas three (1 to 5). Ten patients were cured by a single injection. The mean reduction in

size of the lesion (radiological healing) was found to be 76.6% (61.9% to 93.2%) with a

mean clinical response of 84.5% (73.4% to 100%). Recurrence was seen in two patients

(2.8%) within two years of treatment and both were treated successfully by further

sclerotherapy.

Percutaneous sclerotherapy with polidocanol is a safe alternative to conventional

surgery for the treatment of an aneurysmal bone cyst. It can be used at surgically-

inaccessible sites and treatment can be performed on an out-patient basis.

Since its first description by Jaffe and

Lichtenstein

1

in 1942, many cases of aneurys-

mal bone cyst have been reported.

2

The originof the lesion remains controversial.

3-6

The

radiological criteria of the lesion and its char-

acterisation have also been extensively de-

scribed and reviewed.

7

An eccentrically lo-

cated, lytic, expansile lesion in the metaphysis

with cortical thinning and a subperiosteal

thin shell of bone is considered to be typical of

an aneurysmal bone cyst. Surgical and adju-

vant treatment have a high rate of complica-

tions.

8-12

In recent years percutaneous sclerotherapy

has emerged as an excellent method of treat-

ment for aneurysmal bone cyst, and obviates

the potential functional disabilities such as

joint stiffness and shortening which are com-

monly seen with other methods of treatment.

We report our experience using a novel

method of sclerotherapy using 3% polidocanol

(hydroxypolyaethoxydodecan (Samarth Life-

sciences Pvt. Ltd., Solan, India); available in 2

ml ampoules; 1 ml = 30 mg of polidocanol)

and describe the clinical and radiological

results.

Patients and Methods

Between July 1997 and December 2004, we

treated 72 patients with an aneurysmal bone

cyst. This study is a combined retrospective

and prospective review of these patients.

There were 46 males and 26 females with a

mean age of 15.6 years (3 to 38). There were

39 lesions in the upper limb, 29 in the lower

limb and four in the axial skeleton (Table I).

The mean follow-up was 34 months (26.5 to

80). No patient was lost to follow-up. Pre-

Table I. Site of distribution of the lesions

Bones affected Number of cases

Clavicle 3

Humerus 30

Radius 4

Ulna 2

Pelvis 3

Spine 1

Femur 14

Tibia 6

Fibula 6

Foot 3

Total 72

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TREATMENT OF ANEURYSMAL BONE CYSTS WITH PERCUTANEOUS SCLEROTHERAPY USING POLIDOCANOL 1213

VOL. 88-B, No. 9, SEPTEMBER 2006

operative evaluation included clinical, radiological and

histopathological assessment. Radiographs were obtained

for all the patients and MR scans for patients with axially-

distributed lesions. A histological diagnosis was obtained

using a percutaneous trephine biopsy. When the tissue was

inadequate for establishing a diagnosis, a repeat trephine

biopsy was obtained. In two patients a definitive diagnosis

was not possible on the trephine biopsy specimen and anopen biopsy was performed. All cases of secondary aneu-

rysmal bone cysts were excluded from the study.

Treatment started three months after the biopsy, to give

enough time for spontaneous healing to occur following

bone puncture as has been reported previously.

13

The vol-

ume of the cyst was measured pre-operatively using plain

radiography in two planes by multiplying the length and

the width of the cyst on the anteroposterior view and the

depth on the lateral view. A magnification factor of 10%

was incorporated in the calculations. This calculation was

found to be suitable only for small and spherical lesions less

than 3 cm in diameter.Treatment was undertaken under general anaesthesia for

patients less than 15 years of age and using 2% xylocaine

local anaesthetic with sedation in older patients. Polidoca-

nol was injected into the lesion under fluoroscopic guidance

using a bone-marrow aspiration needle (Gallini Medical

Devices, Mirandula, Italy) or a 16G needle (Gallini Medical

Devices). Approximately 1 ml of 3% polidocanol was

injected per 1 cm

3

volume of the lesion. Back flow of the

sclerosant was prevented by locking the needle for one

minute and subsequently flushing it with 0.5 ml of normal

saline. No more than 10 ml of sclerosant was injected into

any lesion.

Larger and irregular cysts may be measured using ultra-sonography and complex mathematical calculations, but

this was not considered to be necessary in view of the lim-

itation of the amount of drug that could be injected into

these lesions. Subsequent assessment was undertaken at six

and 12 weeks and six-monthly intervals thereafter for up to

approximately seven years. The end-point of treatment was

defined as the time at which the pain had resolved, the cor-

tical thickness of the wall of the cyst had started reforming

and the lesion had stopped growing in size. A second injec-

tion of sclerosant was given if any one or a combination of

the above three parameters was not observed in the first

three months after treatment. Patients were advised to

avoid contact sports and strenuous activity until the lesion

healed. One patient with a lesion in the neck of femur

remained non-weight-bearing for six weeks. Two patients

with an extensive humeral lesion used removable splints

until the completion of treatment.

At each follow-up visit, the lesions were graded radiolog-

ically, by our own system, as grade I (residual lesion < 25%

of the initial lesion), grade II (residual lesion 25% to 49%),

grade III (residual lesion 50% to 74%), and grade IV (resid-

ual lesion 75% or more). The acceptability of treatment,

the occurrence of complications and a functional assess-

ment were also recorded at each follow-up visit according

to the method of Enneking et al.

14

Statistical analysis. We used the Fisher’s exact test, the chi-

square test and Student’s t

-test to analyse our results. A

p value < 0.05 was considered significant.

Results

The results are summarised in Table II.

The mean number of injections was 3 (1 to 5) per patient,

only one being required in ten patients. The mean length of

treatment was 11.9 months (6 to 18). The overall mean

residual size of the lesion at final follow-up was 23.3%

(6.8% to 39.1%). Grade-I healing was found in 48 patients

(66.7%), grade-II in 22 (30.5%) and grade-III in two

patients (2.8%) (Figs 1 to 3). The mean overall rating was

Fig. 1a

Radiographs showing an aneurysmal bone cyst of the proximal humerusa) before treatment and b) one year after sclerotherapy.

Fig. 1b

Table II. Clinical details and results

Parameters Values

Mean age in yrs (range) 15.6 (3 to 38)

Gender (M:F) 46:26

Mean functional score* at the end of treatment (range) 21.8 (10 to 26)

Mean functional score

*

at the end of follow-up (range) 24.3 (12 to 30)Mean number of injections (range) 3 (1 to 5)

Mean length of treatment in mths (range) 11.9 (6 to 18)

Mean volume of lesion in cm3 pre-operatively (range) 26.1 (8.2 to 58.2)

Mean volume of lesion in cm3 at end of follow-up(range)

6 (1.2 to 12.5)

Mean residual size of lesion in % (range) 23.3 (6.8 to 39.1)

* Enneking’s method

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1214 S. RASTOGI, M. K. VARSHNEY, V. TRIKHA, S. A. KHAN, B. CHOUDHURY, R. SAFAYA

THE JOURNAL OF BONE AND JOINT SURGERY

Fig. 3c

Radiographs showing an aneurysmal bone cyst of the r ight side of the pubic ramus a) before treatment and b) 29 months aftersclerotherapy, and CT scans c) before treatment and d) 29 months after sclerotherapy showing complete healing of the lesion aswell as new bone formation.

Fig. 2a

Radiographs showing an aneurysmal bone cyst of the lower end of the fibula a) before treatment and b) 26 months after sclerotherapy.

Fig. 3a Fig. 3b

Fig. 3d

Fig. 2b

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TREATMENT OF ANEURYSMAL BONE CYSTS WITH PERCUTANEOUS SCLEROTHERAPY USING POLIDOCANOL 1215

VOL. 88-B, No. 9, SEPTEMBER 2006

75% (60% to 86.7%) at the end of treatment and 84.5%

(73.4% to 100%) at the final follow-up as assessed by the

system of Enneking et al.

14

Improvement in the functional

score correlated positively with the reduction in size of the

lesion.

The functional scores at the end of treatment and at the

last follow-up were compared with a valid n = 72. Using the

comparison for the paired samples (

t

-test) the result wasfound to be highly significant (p = 0.002). The functional

score at the end of the final follow-up (34 months; 26.5 to

80) was significantly better than that at the end of the initial

treatment (11.9 months; 6 to 18).

There was no correlation between the site treated and the

length of treatment, indicating that the outcome is not

influenced by the site of the lesion.

Complications

Local recurrence was seen in two patients (2.8%), at the

end of treatment (mean 11.92 months, 6 to 18). However,

both were successfully treated with a further single injec-tion of sclerosant. Other complications included induration

at the site of injection (18 cases), hypopigmentation (3),

local inflammatory reaction (1), and an episode of dizziness

(1).

Discussion

An aneurysmal bone cyst is a tumour which attracts contro-

versy principally regarding its characterisation

3-5,8,15,16

and

management.

9,10,17

It has been classically considered as an

arteriovenous fistula,

1

but some consider it to be post-

traumatic

3

in origin and others a de-novo

lesion.

4

A venous

impedance aetiology

5

has also been proposed. However,

most consider it to be some type of vascular malformation,justifying the term ‘benign vascular tumour of bone’.

6,8,11,18

It may be a primary

14

lesion or superimposed upon another

lesion.

19,20

Many forms of treatment have been described including

intralesional procedures, radiation therapy, subtotal or total

excision with or without reconstruction, sclerotherapy and

selective/super-selective embolisation.

8-12,17,19-21

It has also

been reported to regress spontaneously

13

after biopsy and

fracture healing. Curettage with or without bone grafting

is considered to be the treatment of choice for these

lesions.

There is, however, a high recurrence rate and the poten-

tial of disturbance of growth when the lesion is near a phy-

seal plate.

8-11

Marcove et al

8

and Cole

10

reported recur-

rence rates of more than 50% after curettage with or

without bone grafting. Schreuder et al

12

reviewed the liter-

ature and found a recurrence rate of 30.8% after curettage

and bone grafting. High-speed burring and cryotherapy

8,12

have been used as adjuvants in an attempt to reduce the

recurrence rate, to as low as 4% according to Schreuder et

al.

12

However, these procedures are associated with haem-

orrhage, particularly in large lesions, incomplete excision

and physeal injury.

8,10,20

Moreover, surgery is not feasible

at all sites due to anatomical constraints. Endoscopic curet-

tage has also been described in a small series of patients but

long-term results are awaited.

17

Although extralesional excision removes the lesion in

toto

, it may involve extensive surgery with prolonged

immobilisation, and may be complicated by the need for

bone grafts and associated, growth plate injury morbid-

ity.

15

Saucerisation of the lesion has similar complica-tions

8,10

although complete healing has been reported.

10

Radiotherapy

8,11,15

has now been abandoned in view of

the large number of complications including malignant

change. Marcove et al

8

reported uncontrolled lesions in 9%

of their patients and the development of a secondary

tumour in one patient after radiotherapy.

Embolisation

18

was used to reduce the intra-operative

blood loss and proved to be an effective method of treat-

ment. A recurrence rate of 10.5% was reported by De

Cristofaro et al.

18

Super selective embolisation

19

of the

feeding vessels widened the application of this method to

surgically-inaccessible regions. Although the procedure isvery effective, not all aneurysmal bone cysts have a major

feeding vessel which can be satisfactorily embolised. Also

ischaemia of vital neural and visceral structures remains a

major concern.

Sclerosants, in general, act by direct damage to the

endothelial lining, triggering a coagulation cascade and

thrombotic occlusion of blood vessels.

22,23

Several scleros-

ing agents have been used, but an alcoholic solution of

Zein

24-27

is the most popular. Polidocanol has been used

safely in the treatment of varicose veins, telengiectases,

venous malformations of the head, neck and limbs

22,23

gastro-oesophageal varices, endoscopic injection of intesti-

nal vascular malformations and hydrocele of the testis.Methylprednisolone acetate,

28

calcitonin and radionuclei-

des have also been used sporadically.

To our knowledge, the use of polidocanol for the treat-

ment of aneurysmal bone cysts has been described in only

one previous study.

29

We used polidocanol because of its

proven efficacy in the treatment of vascular malformations

at various sites with relatively few complications.

22,23

The

result was satisfactory in more than 97% (70) of our

patients. The residual lesion was on average only 23.3% of

the initial lesion. The rating of the clinical response was

75% at the end of treatment which improved to 84.5% at

the final follow-up. This is comparable to the value of 90%

(63% to 100%) observed by Marcove et al

8

who used

curettage and cryotherapy. A mean of three injections (1 to

5) was required. Clinical and radiological improvement

continued after the completion of treatment suggesting an

ongoing healing process. A recurrence occurred in only two

patients (2.8%), both occurring within two years of the end

of treatment. Both were successfully treated by further

sclerotherapy.

We found the method to be safe and effective with mini-

mal complications. The response to therapy is graded, eas-

ily observed and followed. Patients with grade-II healing do

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1216 S. RASTOGI, M. K. VARSHNEY, V. TRIKHA, S. A. KHAN, B. CHOUDHURY, R. SAFAYA

THE JOURNAL OF BONE AND JOINT SURGERY

not require long follow-up as most are healed by one year

after treatment. Patient acceptance is very good as the pro-

cedure respects cosmesis and can be done on an out-patient

basis.

Potential complications with the use of polidocanol

include hypopigmentation,

30

necrosis at the site of the

injection in the case of extravasation, pulmonary embo-

lism, osteomyelitis, allergic reactions and anaesthetic com-plications.

22,23

However, none of these apart from

hypopigmentation, which subsided spontaneously, was

encountered in our study.

Policodanol is relatively contraindicated for patients

with skin conditions at the site of injection, heart disease,

asthma and pregnancy, especially in the first trimester.

22,23

It should not be injected intra-arterially because severe

necrosis can occur,

23

and it should not be used if the lesion

causes neurovascular compression requiring surgical treat-

ment. We did not use it at any intracranial site.

We recommend that sclerotherapy should be carried out

only after an appropriate assessment of the patient underaseptic conditions and fluoroscopic guidance. Prevention of

extravasation is the key to avoiding complications. Follow-

up of at least two years is recommended. Recurrences may

take place within two years.

Sclerotherapy using policodanol for the treatment of pri-

mary aneurysmal bone cyst could prove to be a highly effec-

tive and acceptable alternative to conventional surgical

techniques in view of its safety and efficacy.

No benefits in any form have been received or will be received from a commer-

cial party related directly or indirectly to the subject of this article.

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