Clinical Paper

Clinical Pathology

Int. J. Oral Maxillofac. Surg. 2009; 38: 671–676doi:10.1016/j.ijom.2009.01.016, available online at http://www.sciencedirect.com

Treatment of large recurrentaneurysmal bone cysts ofmandible: transosseousintralesional embolization as anadjunct to resectionV. V. Kumar, N. A. Malik, D. B. Kumar: Treatment of large recurrent aneurysmalbone cysts of mandible: transosseous intralesional embolization as an adjunct toresection. Int. J. Oral Maxillofac. Surg. 2009; 38: 671–676. # 2009 InternationalAssociation of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rightsreserved.

Abstract. The aneurysmal bone cyst is an uncommon, but well described, bone lesionoccurring most commonly in long bones, the pelvis and vertebrae. It is relativelyrare in the maxillofacial region. Various treatment options have been proposed. Theauthors present three cases of recurrent, large aneurysmal bone cysts of themandible successfully treated by en block resection and reconstruction using freefibula flap following diagnostic arteriography and preoperative transcutaneousintralesional embolization. Preoperative embolization produced a relatively blood-less field from which the tumour could be completely excised.

0901-5027/060671 + 06 $36.00/0 # 2009 International Association of Oral and Maxillofacial Surgeo

V. V. Kumar, N. A. Malik,D. B. KumarDepartment of Oral and Maxillofacial Surgery,Nair Hospital Dental College, Mumbai, India

Keywords: aneurysmal bone cyst; en blockresection; embolization; selective arteriogra-phy.

Accepted for publication 29 January 2009Available online 24 February 2009

Aneurysmal bone cysts (ABCs) are benign,non-neoplastic, expansile, vascular locallydestructive lesions. WHO classifies ABC asa tumour-like lesion and defines it as ‘anexpanding osteolytic lesion consisting ofblood-filled spaces of variable size sepa-rated by connective tissue septa containingtrabeculae of osteoid tissue and osteoclastgiant cells’19. JAFFE and LICHTEN-STEIN10 recognized it as a distinct entityin 1942 with the word ‘aneurysmal’ toemphasize the ‘blown out’, distended con-

tour of the affected bone, and the words‘bone cyst’ to underscore that when thelesion is entered through a thin shell ofbone, it appears largely as a blood-filledcavity. ABCs are most frequently seen inthe long bones (50%) and vertebrae (20%).They are relatively rare in the jaw, account-ing for about 1.9% of all ABCs of theskeleton, and about 1.5% of all nonodonto-genic cysts of the jaw15.

Various treatment options have beendescribed for ABCs of which curettage

and en bloc excision are the methods ofchoice. Other modalities include radiation,cryotherapy, percutaneous intralesionalinjection, calcitonin therapy and emboli-zation.

There are reports of embolotherapy (asan adjunct to surgical treatment or asa definitive treatment modality) beingused to treat ABCs of the spine, longbones and pelvis. Intralesional embo-lotherapy has been used as a definitivetreatment wherein the lesions show com-

ns. Published by Elsevier Ltd. All rights reserved.

672 Kumar et al.

Fig. 1. Patient no.1. (A): preoperative frontal photograph. (B): 3D CT scan showing multipleperforations of buccal and lingual cortices. (C): final glue cast showing glue occupying thelesion. (D): panoramic radiograph showing fibula flap reconstruction of the mandible. (E):postoperative frontal profile photograph. (F): SPECT scan suggesting increased radiotraceruptake at the reconstruction site.

plete involution following emboliza-tion12; or as an adjunct treatment whereinembolotherapy limits blood loss duringsurgery4. There is no literature regardingthe use of this treatment modality formandibular lesions. The authors presentthree cases of large recurrent aneurysmalbone cysts of the mandible successfullytreated by en block resection and recon-struction using free fibula graft followingdiagnostic arteriography and preopera-tive transcutaneous intralesional emboli-zation.

Patients and methods

Between January 2004 and May 2007, sixpatients aged 8–32 years (mean, 20.33years) presented with a diagnosis ofABC; two cases were in the maxilla andfour in the mandible.

Three patients had recurrent largelesions (more than 7 cm in maximaldimension) with multiple perforationsof both the lingual and buccal corticesof the mandible. The patients had under-gone surgery in other institutions and hadbeen referred following recurrence andsudden enlargement of the swelling. Nodetails of the attempted surgical pro-cedures were available. Preoperativeimaging modalities included an orthopan-tamogram, a CT scan, technetium per-technetate bone scintigraphy (to rule outmultiple skeletal involvements) andmagnetic resonance imaging (MRI; foracademic purposes). After examination,a biopsy was taken, which suggestedABC. Only these patients were chosenfor the study. Informed consent wasobtained from all the patients (and parentsin cases of minors) after the risks andbenefits of the treatment were fullyexplained. In these patients, the treatmentprotocol consisted of a first surgical pro-cedure for biopsy under local anaesthesiaconfirming the lesion as an ABC; a secondprocedure including angiography and per-cutaneous embolotherapy under generalanaesthesia; and a final surgical procedureinvolving resection and primary recon-struction using free fibula flap under gen-eral anaesthesia.

The three patients presented with swel-ling, pain and mobility of the teeth relatedto the bony expansion. All the lesions werepresent in the angle of the mandible region(Fig. 1) with varying degrees of condylar,ramal and body involvement. One patient(patient no. 2) had involvement of thecoronoid process (Fig. 2). In one patient(patient no. 3) the lesion was on the rightside of the mandible (Fig. 3) and on the leftside in the other two (patient nos. 1 and 2).

All patients had an angiography viaa transfemoral approach under generalanaesthesia. Their external carotid angio-grams revealed stretching of the facialartery and its displacement laterally andposteriorly (Fig. 4). No vascularity toany of the lesions was evident. A deci-sion was taken to approach the lesionsthrough transcutaneous, direct intraos-seous puncture. Multiple18 gauge nee-dles were inserted transcutaneously into

the lesion under constant fluoroscopicguidance. When good back-flow of bloodwas seen, diluted n-butyl-cyano acrylateglue (Histoacryl, B-Braun, Tuttlingen,Germany) was injected. Manual com-pression was provided for at least10 min following the procedure. Patientswere prescribed antibiotics, analgesicsfor 1 week and discharged 24 h afterthe procedure. Follow-up consisted ofclinical and radiological examination

Treatment of large recurrent aneurysmal bone cysts of mandible 673

Fig. 2. Patient no.2. (A): preoperative frontal photograph. (B): CT scan coronal section showingextension of the lesion, ballooning of the condylar and coronoid process. (C): final glue castshowing glue occupying the lesion. (D): surgical approach for the resection of the lesion. (E):postoperative frontal profile photograph. (F): panoramic radiograph showing fibula flapreconstruction of the mandible.

(panoramic radiographs, CT scans) 2 and4 months after the procedure.

All the patients showed lack of boneformation 4 months after the procedure(Fig. 3) and so underwent en bloc resec-tion with reconstruction using the freefibula flap with double barrelling in thebody region under general anaesthesia.Patients were followed-up at intervals of1, 2, 4, 6 and 12 months after surgery.

Results

In all the patients, needle aspiration waspositive and dark venous fluid was aspi-rated. Diluted n-butyl cyanoacrylateglue (Histoacryl, B-Braun, Tuttlingen,Germany) was injected successfully. Theamount injected was 20 cc (patient no. 1),21 cc (patient no.2) and 19 cc (patientno.3). Good casts were seen following

the procedure showing that the majorityof the lesions were occupied by glue.Fluoroscopic guidance ensured that theinjection was confined to the lesion. Noneof the patients had severe complicationsand pain was reported as the main com-plaint. Pain was most severe during thefirst 48 h and decreased thereafter; it waswell controlled by analgesia and bed rest.Four months after the procedure, none ofthe patients showed any radiological signsof involution so they were considered foren bloc resection. Gross examination ofthe resected specimen showed glue in thelesion (Fig. 4). There was minimal bloodloss during surgery. The free-flap wassuccessful in all the patients, with SPECTscan showing increased radiotracer uptakeat the reconstruction site. Follow-up at 1and 3 years showed no recurrence. All thepatients had good oral function and nodonor site morbidity following surgery.

Discussion

Although ABC is a benign lesion, it canbehave in a locally aggressive manner,because of its rapid growth and osteolyticcapacity. LICHTENSTEIN12,13 suggestedthat ABCs may be a result of alterations inthe local haemodynamics that may be akinto a periosteal or intraosseous malforma-tion, a concept supported by MIRRA14

based on histological and radiologicalsimilarities between ABCs and soft tissuemalformations.

Histological findings such as blood fill-ing, anastomosis, and cavernous spacesseparated by septa (Fig. 4) are character-istic of both ABCs and soft-tissue venousmalformations17. MRI and angiographydemonstrate similar findings in these enti-ties: lobulated contours and internal septa,with low signal intensity on T1-weightedMRI (Fig. 4) and, in particular, very highsignal intensity on T2-weighted MRI.Fluid levels often occur in both entitiesand reflect low intralesional blood flow,which allows differentiation betweendependent and nondependent blood com-ponents. Angiographic findings are typicalin most cases of low intralesional bloodflow lesions, with no, or discrete, arterialhypervascularity, but there may bedelayed and prolonged lesion staining inthe venous phase. These findings were alsoseen in the authors’ patients, whichshowed no direct vascularity to the lesion(Fig. 4).

Various treatments have been reportedfor ABCs, including observation and long-term follow-up, which may lead tospontaneous regression8. Percutaneousinjection of a fibrosing agent (Ethibloc,

674 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 thebody with multiple lingual and buccal perforations. (B): final glue cast showing glue occupying the lesion. (C): axial section of CT scan taken 4months after embolization does not show signs of resolution of the lesion. (D): panoramic radiograph showing fibula flap reconstruction of themandible.

Ethicon) has also been employed withgood results1.

Intralesional calcitonin18 injections incombination with methylprednisolone5

have been used, but this therapy oftentakes a long time with repeated multipleinjections and the response is unpredict-able. The injections are thought to com-bine the inhibitory angiostatic andfibroblastic effects of methylprednisolonewith the osteoclastic inhibitory effect andtrabecular bone-stimulating properties ofcalcitonin.

Surgical curettage is the most commonform of treatment for this lesion. Therecurrence rate (most likely in the first 2years after surgery) is 10–54%3,22 in casesof extra-gnathic lesions. The recurrencerate after curettage in the jaws ranges from016 to 53%20. Many authors attribute thelarge recurrence rates to incompleteremoval during surgery20. A problem thatmight lead to incomplete removal is themassive haemorrhage that may be encoun-tered, which might require ligation of theexternal carotid artery as a precautionarymeasure6. Although cryotherapy2 andradiotherapy23 have supplemented curet-tage to decrease the recurrence rate, theuse of the latter is strongly discouraged as

it is likely to induce sarcomatous changein the irradiated bone21. The treatmentmodality most likely to effect a completecure is en bloc resection20, but this isrestricted to large and recurrent lesionsowing to the morbidity of the procedure.

In the present case, there were manyreasons for using adjunctive embolother-apy. Numerous successful cases usingarterial embolization, as an adjunct tosurgery or as a sole therapy, have beenreported particularly in the pelvis24, longbones4 and spine11. Arterial embolizationwas first used preoperatively to decreasevascularity and intraoperative haemor-rhage. It has been reported that embo-lotherapy occludes vascularity of thelesion without interfering with the vascu-larity of the surrounding tissues, whichmay lead to involution of the soft tissuecomponent. The response of ABCs toembolization has been involution of thesoft tissue component, sclerosis andossification4,11,24. This mineralizationbecomes apparent two or more monthsafter embolization. In some of the extra-gnathic cases reported, significant invo-lution of the soft tissue mass withaugmented ossification occurred so thatsurgery was avoided. It has been sug-

gested that sclerotherapy should havethe same efficacy in ABCs as in soft tissuevenous malformations because theseentities represent similar processes invol-ving bone and soft tissue, respectively7.Another reason for using embolotherapyis that it would reduce bleeding from thelesion while removing it. This wouldeliminate the need for multiple bloodtransfusions and result in a clearer surgi-cal field allowing complete removal of thelesion, reducing recurrence6,20.

All the cases treated by the authors werereferrals following attempted curettage.They were treated as recurrent lesions,though the extent of curettage carriedout was not known to the authors. Allthe cases involved large lesions, withmultiple perforations of the mandible,and erosions and perforations of bothbuccal and lingual cortices, with extre-mely thin inferior borders. It was decidedto treat these lesions by en bloc resection,followed by primary reconstruction usingthe free fibula flap because it is consideredthe gold standard for the reconstruction ofmandibular defects9.

The lesions did not resolve on emboli-zation, probably because they were largeand had multiple perforations. Emboliza-

Treatment of large recurrent aneurysmal bone cysts of mandible 675

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

tion did allow safe resection in a relati-vely bloodless field. This ensured com-plete removal of the lesion, removing thechance of recurrence. Embolization seemsto be a useful procedure in the treatment ofABCs and could be tried as the primarytreatment modality, but further studies areneeded before it becomes the standardprotocol for the treatment of these uncom-mon lesions.

Funding

None declared.

Competing interests

None.

Ethical approval

Not required.

Acknowledgements. The authors wouldlike to acknowledge: Dr. AMRESH S.BALIARSING, Professor and Head,Department of Plastic and Reconstructive

Surgery, King Edward Memorial Hospital,Parel, Mumbai, India for carrying out thereconstructive procedures.

We would also like to thank Dr UdayLimaye and Dr Manish Kumar Srivastava,Department of Interventional Neurora-diology, King Edward Memorial Hospital,Parel, Mumbai, India for carrying outthe arteriography and embolization proce-dures.

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Address: Vinay V. Kumar5-CCheloor TowersPootholeThrissurKeralaIndiaPin: 680004E-mail: vinay_maxfax@yahoo.co.in