YIJOM-1524; No of Pages 6

Int. J. Oral Maxillofac. Surg. 2009; xxx: xxx–xxx

doi:10.1016/j.ijom.2009.01.016, available online at http://www.sciencedirect.com

Clinical Paper
Clinical Pathology

Treatment of large recurrent V. V. Kumar, N. A. Malik,

D. B. Kumar
Department of Oral and Maxillofacial Surgery,

aneurysmal bone cysts of

Nair Hospital Dental College, Mumbai, India

mandible: transosseous
intralesional embolization as an
adjunct to resection
V. V. Kumar, N. A. Malik, D. B. Kumar: Treatment of large recurrent aneurysmal
bone cysts of mandible: transosseous intralesional embolization as an adjunct to
resection. Int. J. Oral Maxillofac. Surg. 2009; xxx: xxx–xxx. # 2009 International
Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights
reserved.

Abstract. The aneurysmal bone cyst is an uncommon, but well described, bone lesion
occurring most commonly in long bones, the pelvis and vertebrae. It is relatively
rare in the maxillofacial region. Various treatment options have been proposed. The
authors present three cases of recurrent, large aneurysmal bone cysts of the
Keywords: aneurysmal bone cyst; en block
mandible successfully treated by en block resection and reconstruction using free resection; embolization; selective arteriogra-
fibula flap following diagnostic arteriography and preoperative transcutaneous phy.
intralesional embolization. Preoperative embolization produced a relatively blood-
less field from which the tumour could be completely excised. Accepted for publication 29 January 2009

Aneurysmal bone cysts (ABCs) are benign, tour of the affected bone, and the words and en bloc excision are the methods of
non-neoplastic, expansile, vascular locally ‘bone cyst’ to underscore that when the choice. Other modalities include radiation,
destructive lesions. WHO classifies ABC as lesion is entered through a thin shell of cryotherapy, percutaneous intralesional
a tumour-like lesion and defines it as ‘an bone, it appears largely as a blood-filled injection, calcitonin therapy and emboli-
expanding osteolytic lesion consisting of cavity. ABCs are most frequently seen in zation.
blood-filled spaces of variable size sepa- the long bones (50%) and vertebrae (20%). There are reports of embolotherapy (as
rated by connective tissue septa containing They are relatively rare in the jaw, account- an adjunct to surgical treatment or as
trabeculae of osteoid tissue and osteoclast ing for about 1.9% of all ABCs of the a definitive treatment modality) being
giant cells’19. JAFFE and LICHTEN- skeleton, and about 1.5% of all nonodonto- used to treat ABCs of the spine, long
STEIN10 recognized it as a distinct entity genic cysts of the jaw15. bones and pelvis. Intralesional embo-
in 1942 with the word ‘aneurysmal’ to Various treatment options have been lotherapy has been used as a definitive
emphasize the ‘blown out’, distended con- described for ABCs of which curettage treatment wherein the lesions show com-

0901-5027/000001+06 $30.00/0 # 2009 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: V.V.. Kumar, et al., Treatment of large recurrent aneurysmal bone cysts of mandible: transosseous
intralesional embolization as an adjunct to resection, Int J Oral Maxillofac Surg (2009),
YIJOM-1524; No of Pages 6

2 Kumar et al.

plete involution following emboliza-

tion12; or as an adjunct treatment wherein
embolotherapy limits blood loss during
surgery4. There is no literature regarding
the use of this treatment modality for
mandibular lesions. The authors present
three cases of large recurrent aneurysmal
bone cysts of the mandible successfully
treated by en block resection and recon-
struction using free fibula graft following
diagnostic arteriography and preopera-
tive transcutaneous intralesional emboli-
zation.

Patients and methods

Between January 2004 and May 2007, six
patients aged 8–32 years (mean, 20.33
years) presented with a diagnosis of
ABC; two cases were in the maxilla and
four in the mandible.
Three patients had recurrent large
lesions (more than 7 cm in maximal
dimension) with multiple perforations
of both the lingual and buccal cortices
of the mandible. The patients had under-
gone surgery in other institutions and had
been referred following recurrence and
sudden enlargement of the swelling. No
details of the attempted surgical pro-
cedures were available. Preoperative
imaging modalities included an orthopan-
tamogram, a CT scan, technetium per-
technetate bone scintigraphy (to rule out
multiple skeletal involvements) and
magnetic resonance imaging (MRI; for
academic purposes). After examination,
a biopsy was taken, which suggested
ABC. Only these patients were chosen
for the study. Informed consent was
obtained from all the patients (and parents
in cases of minors) after the risks and
benefits of the treatment were fully
explained. In these patients, the treatment
protocol consisted of a first surgical pro-
cedure for biopsy under local anaesthesia
confirming the lesion as an ABC; a second
procedure including angiography and per- Fig. 1. Patient no.1. (A): preoperative frontal photograph. (B): 3D CT scan showing multiple
cutaneous embolotherapy under general perforations of buccal and lingual cortices. (C): final glue cast showing glue occupying the
anaesthesia; and a final surgical procedure lesion. (D): panoramic radiograph showing fibula flap reconstruction of the mandible. (E):
involving resection and primary recon- postoperative frontal profile photograph. (F): SPECT scan suggesting increased radiotracer
struction using free fibula flap under gen- uptake at the reconstruction site.
eral anaesthesia.
The three patients presented with swel- All patients had an angiography via the lesion under constant fluoroscopic
ling, pain and mobility of the teeth related a transfemoral approach under general guidance. When good back-flow of blood
to the bony expansion. All the lesions were anaesthesia. Their external carotid angio- was seen, diluted n-butyl-cyano acrylate
present in the angle of the mandible region grams revealed stretching of the facial glue (Histoacryl, B-Braun, Tuttlingen,
(Fig. 1) with varying degrees of condylar, artery and its displacement laterally and Germany) was injected. Manual com-
ramal and body involvement. One patient posteriorly (Fig. 4). No vascularity to pression was provided for at least
(patient no. 2) had involvement of the any of the lesions was evident. A deci- 10 min following the procedure. Patients
coronoid process (Fig. 2). In one patient sion was taken to approach the lesions were prescribed antibiotics, analgesics
(patient no. 3) the lesion was on the right through transcutaneous, direct intraos- for 1 week and discharged 24 h after
side of the mandible (Fig. 3) and on the left seous puncture. Multiple18 gauge nee- the procedure. Follow-up consisted of
side in the other two (patient nos. 1 and 2). dles were inserted transcutaneously into clinical and radiological examination

Please cite this article in press as: V.V.. Kumar, et al., Treatment of large recurrent aneurysmal bone cysts of mandible: transosseous
intralesional embolization as an adjunct to resection, Int J Oral Maxillofac Surg (2009),
YIJOM-1524; No of Pages 6

Treatment of large recurrent aneurysmal bone cysts of mandible 3

the procedure showing that the majority

of the lesions were occupied by glue.
Fluoroscopic guidance ensured that the
injection was confined to the lesion. None
of the patients had severe complications
and pain was reported as the main com-
plaint. Pain was most severe during the
first 48 h and decreased thereafter; it was
well controlled by analgesia and bed rest.
Four months after the procedure, none of
the patients showed any radiological signs
of involution so they were considered for
en bloc resection. Gross examination of
the resected specimen showed glue in the
lesion (Fig. 4). There was minimal blood
loss during surgery. The free-flap was
successful in all the patients, with SPECT
scan showing increased radiotracer uptake
at the reconstruction site. Follow-up at 1
and 3 years showed no recurrence. All the
patients had good oral function and no
donor site morbidity following surgery.

Discussion
Although ABC is a benign lesion, it can
behave in a locally aggressive manner,
because of its rapid growth and osteolytic
capacity. LICHTENSTEIN12,13 suggested
that ABCs may be a result of alterations in
the local haemodynamics that may be akin
to a periosteal or intraosseous malforma-
tion, a concept supported by MIRRA14
based on histological and radiological
similarities between ABCs and soft tissue
malformations.
Histological findings such as blood fill-
ing, anastomosis, and cavernous spaces
separated by septa (Fig. 4) are character-
istic of both ABCs and soft-tissue venous
malformations17. MRI and angiography
demonstrate similar findings in these enti-
ties: lobulated contours and internal septa,
with low signal intensity on T1-weighted
MRI (Fig. 4) and, in particular, very high
signal intensity on T2-weighted MRI.
Fluid levels often occur in both entities
and reflect low intralesional blood flow,
Fig. 2. Patient no.2. (A): preoperative frontal photograph. (B): CT scan coronal section showing which allows differentiation between
extension of the lesion, ballooning of the condylar and coronoid process. (C): final glue cast dependent and nondependent blood com-
showing glue occupying the lesion. (D): surgical approach for the resection of the lesion. (E): ponents. Angiographic findings are typical
postoperative frontal profile photograph. (F): panoramic radiograph showing fibula flap in most cases of low intralesional blood
reconstruction of the mandible.
flow lesions, with no, or discrete, arterial
hypervascularity, but there may be
(panoramic radiographs, CT scans) 2 and Results delayed and prolonged lesion staining in
4 months after the procedure. the venous phase. These findings were also
All the patients showed lack of bone In all the patients, needle aspiration was seen in the authors’ patients, which
formation 4 months after the procedure positive and dark venous fluid was aspi- showed no direct vascularity to the lesion
(Fig. 3) and so underwent en bloc resec- rated. Diluted n-butyl cyanoacrylate (Fig. 4).
tion with reconstruction using the free glue (Histoacryl, B-Braun, Tuttlingen, Various treatments have been reported
fibula flap with double barrelling in the Germany) was injected successfully. The for ABCs, including observation and long-
body region under general anaesthesia. amount injected was 20 cc (patient no. 1), term follow-up, which may lead to
Patients were followed-up at intervals of 21 cc (patient no.2) and 19 cc (patient spontaneous regression8. Percutaneous
1, 2, 4, 6 and 12 months after surgery. no.3). Good casts were seen following injection of a fibrosing agent (Ethibloc,

Please cite this article in press as: V.V.. Kumar, et al., Treatment of large recurrent aneurysmal bone cysts of mandible: transosseous
intralesional embolization as an adjunct to resection, Int J Oral Maxillofac Surg (2009),
YIJOM-1524; No of Pages 6

4 Kumar et al.

Fig. 3. Patient no. 3. (A): preoperative axial section of CT scan revealing a multilocular lesion extending from the angle of the mandible to the
body with multiple lingual and buccal perforations. (B): final glue cast showing glue occupying the lesion. (C): axial section of CT scan taken 4
months after embolization does not show signs of resolution of the lesion. (D): panoramic radiograph showing fibula flap reconstruction of the
mandible.

Ethicon) has also been employed with it is likely to induce sarcomatous change gested that sclerotherapy should have
good results1. in the irradiated bone21. The treatment the same efficacy in ABCs as in soft tissue
Intralesional calcitonin18 injections in modality most likely to effect a complete venous malformations because these
combination with methylprednisolone5 cure is en bloc resection20, but this is entities represent similar processes invol-
have been used, but this therapy often restricted to large and recurrent lesions ving bone and soft tissue, respectively7.
takes a long time with repeated multiple owing to the morbidity of the procedure. Another reason for using embolotherapy
injections and the response is unpredict- In the present case, there were many is that it would reduce bleeding from the
able. The injections are thought to com- reasons for using adjunctive embolother- lesion while removing it. This would
bine the inhibitory angiostatic and apy. Numerous successful cases using eliminate the need for multiple blood
fibroblastic effects of methylprednisolone arterial embolization, as an adjunct to transfusions and result in a clearer surgi-
with the osteoclastic inhibitory effect and surgery or as a sole therapy, have been cal field allowing complete removal of the
trabecular bone-stimulating properties of reported particularly in the pelvis24, long lesion, reducing recurrence6,20.
calcitonin. bones4 and spine11. Arterial embolization All the cases treated by the authors were
Surgical curettage is the most common was first used preoperatively to decrease referrals following attempted curettage.
form of treatment for this lesion. The vascularity and intraoperative haemor- They were treated as recurrent lesions,
recurrence rate (most likely in the first 2 rhage. It has been reported that embo- though the extent of curettage carried
years after surgery) is 10–54%3,22 in cases lotherapy occludes vascularity of the out was not known to the authors. All
of extra-gnathic lesions. The recurrence lesion without interfering with the vascu- the cases involved large lesions, with
rate after curettage in the jaws ranges from larity of the surrounding tissues, which multiple perforations of the mandible,
016 to 53%20. Many authors attribute the may lead to involution of the soft tissue and erosions and perforations of both
large recurrence rates to incomplete component. The response of ABCs to buccal and lingual cortices, with extre-
removal during surgery20. A problem that embolization has been involution of the mely thin inferior borders. It was decided
might lead to incomplete removal is the soft tissue component, sclerosis and to treat these lesions by en bloc resection,
massive haemorrhage that may be encoun- ossification4,11,24. This mineralization followed by primary reconstruction using
tered, which might require ligation of the becomes apparent two or more months the free fibula flap because it is considered
external carotid artery as a precautionary after embolization. In some of the extra- the gold standard for the reconstruction of
measure6. Although cryotherapy2 and gnathic cases reported, significant invo- mandibular defects9.
radiotherapy23 have supplemented curet- lution of the soft tissue mass with The lesions did not resolve on emboli-
tage to decrease the recurrence rate, the augmented ossification occurred so that zation, probably because they were large
use of the latter is strongly discouraged as surgery was avoided. It has been sug- and had multiple perforations. Emboliza-

Please cite this article in press as: V.V.. Kumar, et al., Treatment of large recurrent aneurysmal bone cysts of mandible: transosseous
intralesional embolization as an adjunct to resection, Int J Oral Maxillofac Surg (2009),
YIJOM-1524; No of Pages 6

Treatment of large recurrent aneurysmal bone cysts of mandible 5

Fig. 4. (A): T1-weighted MRI showing hypodense cystic areas separated by septae. (B): external carotid angiography revealing stretching of the
facial artery with no vascularity of the lesion. (C): histopathology showing cavernous spaces separated by septae. (D): photograph of the resected
specimen showing glue (arrows) in the lesion.

tion did allow safe resection in a relati- Surgery, King Edward Memorial Hospital, ging findings and embolotherapy. Am J
vely bloodless field. This ensured com- Parel, Mumbai, India for carrying out the Roentgenol 1989: 153: 369–373.
plete removal of the lesion, removing the reconstructive procedures. 5. Gladden Jr ML, Gillingham BL,
chance of recurrence. Embolization seems Hennrikus W. Aneurysmal bone cyst
We would also like to thank Dr Uday of the first cervical vertebrae in a child
to be a useful procedure in the treatment of treated with percutaneous intralesional
Limaye and Dr Manish Kumar Srivastava,
ABCs and could be tried as the primary injection of calcitonin and methylpredni-
Department of Interventional Neurora-
treatment modality, but further studies are solone. A case report. Spine 2000: 25:
diology, King Edward Memorial Hospital,
needed before it becomes the standard 527–530 discussion 531.
Parel, Mumbai, India for carrying out
protocol for the treatment of these uncom- 6. Gruskin SE, Dahlin DC. Aneurysmal
the arteriography and embolization proce-
mon lesions. bone cysts of the jaws. J Oral Surg 1968:
dures. 26: 523–528.
7. Guibaud L, Herbreteau D, Dubois J,
Funding Stempfle N, Berard J, Pracros JP,
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Please cite this article in press as: V.V.. Kumar, et al., Treatment of large recurrent aneurysmal bone cysts of mandible: transosseous
intralesional embolization as an adjunct to resection, Int J Oral Maxillofac Surg (2009),