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Case Study Open Access

Aneurysmal Bone Cyst located in the Mandibular Condyle
Sandro Pelo*, Giulio Gasparini, Roberto Boniello, Alessandro Moro and
Pier Francesco Amoroso*

Address: Department of Oral and Maxillofacial Surgery, Catholic University of S. Cuore, A. Gemelli Rome. Via G. Moscati 31/33 00168 Rome Italy
Email: Sandro Pelo* - [email protected]; Giulio Gasparini - [email protected]; Roberto Boniello - [email protected];
Alessandro Moro - [email protected]; Pier Francesco Amoroso* - [email protected]
* Corresponding authors

Published: 16 February 2009 Received: 24 January 2008

Accepted: 16 February 2009
Head & Face Medicine 2009, 5:8 doi:10.1186/1746-160X-5-8
This article is available from: http://www.head-face-med.com/content/5/1/8
© 2009 Pelo et al; licensee BioMed Central Ltd.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0),
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract
A rare case of aneurysmal bone cyst (ABC) located in the mandibular condyle in a 10-year-old boy
is presented. The patient came to our attention for a sudden swelling in the right
temporomandibular region, the mouth opening was not reduced.
A rapid growing mass, depicting soft tissue invasion, in the right condyle of the mandible was found.
Clinically and radiographically it resembled to a malignant lesion. The surgical excision of the
mandibular condyle allowed a complete removal of the lesion. The histological examination
revealed a pseudocystic expanding osteolytic lesion containing blood-filled space separated by
connective tissue and many osteoclastic giant cells, which was a conventional vascular ABC.
The ABC is an infrequent bone lesion which can only be found very rarely at the craniofacial
skeleton. There have been described about 160 cases of ABC originated in the molar region or in
upper maxilla and even more rare is the location of this cyst in the mandibular condyle. Only 6
cases were reported in the literature to date.
A complete surgical resection of this osteolytic lesion is the treatment of choice considering its high
recurrence rate. The condyle was not replaced with any graft. Therefore a functional device was
used after surgery to overcome the lack of the condyle and to stimulate the growth of the ramus.

Introduction the ABC is extremely rare with 0.5%. The average age of
The aneurysmal bone cysts are benign neoformations occurrence is 13 years and 80% of patients are less than 20
which can affect all the skeleton bones. More than half years old with no gender predilection [1,5]. An aneurys-
occur in the metaphysic of long bones (especially femur mal bone cyst of the condyle is even more unusual, with
and tibia) and between 12 and 30% in the spine. ABC only 6 cases reported in the literature to date [[6-10], and
occurs very rarely in the jaws, about 160 cases have been [11]].
reported and two thirds were located in mandible (the
body of the mandible 40%, the ramus 30% and the angle The aneurysmal bone cyst of the jaw is a pseudocystic
19%) and one third in the maxilla [[1-3] and [4]]. They lesion. It is a rapidly growing and destructive bone lesion
represent about 1.5% of all nonodontogenic and nonepi- characterized by replacement of the normal bone with
thelial cystic of the jaws. Considering all types of jaw cysts fibro-osseous tissue containing blood-filled sinusoidal or

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cavernous spaces. However an unusual solid, focally hem-

orrhagic variant has recently been reported [2]. The solid
type (5% of the cases) may present as a small, asympto-
matic lesion first noticed as a radiolucency on a routine
radiograph, or a small swelling. Bleeding is not as brisk
during surgery with this type of ABC as with the vascular
type. The other extreme is the vascular type of ABC (95%
of the cases), which manifests as a rapidly growing, expan-
sive, destructive lesion causing cortical perforation and
soft tissue invasion. A third form, or "mixed" variant,
demonstrates features of both the vascular and solid
types. In fact, this may be a transitory phase of the lesion
because sudden "activation" or rapid enlargement of sta-
ble lesions has been reported [[6,9] and [10]].

The etiology of ABC is unclear and controversial. One the-

ory states that trauma causes an inciting injury to perio-
steal vessels, thus initiating the development of ABC [12].
However Tillman et al, reporting 95 cases, demonstrated
no significant history of antecedent trauma [4]. Jaffé and
Lichtenstein refer to alteration in local haemodynamics
causing increased venous pressures and engorgement of
the vascular bed in the transformed bone, leading to
resorption, connective tissue replacement, and osteoid
formation [13]. Additional theories about the etiology of
this lesion are a subperiosteal intraosseous hematoma
[12] or other publications state that the ABC is a second-
ary phenomena occurring in primary cystic lesions of
bone or tumours [14]. Cyst 1soft tissue
Pre-operative
depicted
Figure imageinvasion
showingofthe
thesevere
vascular
swelling
Aneurismal
which Bone
Pre-operative image showing the severe swelling
Histologically the ABC is considered a pseudocyst due to which depicted soft tissue invasion of the vascular
the absence of an epithelial wall. The ABC is an expanding Aneurismal Bone Cyst. The patient was able to open
osteolytic lesion containing blood-filled spaces of variable widely.
size, separated by connective tissue by bone trabeculae or
osteoid tissue and many osteoclastic giant cells. The lesion
does not have any clinical or radiological specificity. appreciate that the lesion had totally replaced the man-
Recurrence has been reported confirming the aggressive dibular right condyle and part of the mandibular ascend-
behaviour of this lesion [3]. ing ramus. The lesion substituted the bone marrow of
these anatomical structures and the cortical bone was sub-
Case report stantially expanded and perforated. The condyle was so
A 10-year-old male patient presented with a painless mass ballooned that it was evident through extra oral examina-
in the right temporomandibular region. The swelling was tion. Part of the coronoid process was affected by the infil-
evident since one month and it was hard and sensitive to trative osteolytic process [Figures 3, 4 and 5]. The
palpation. The patient was able to open widely; the man- magnetic resonant imaging (MRI) demonstrated a high-
dible could move in all axes without any limitation. The signal intensity within the lesion itself and a low-signal at
lesion caused facial asymmetry. There was no paresthesia the periphery [Figure 6]. The clinical presentation and the
in the area innervated by the right mandibular nerve or by radiographic appearance of this lesion could have been
the right facial nerve. The boy did not have any previous associated to osteosarcoma, ameloblastma, myxoma or
trauma in the swollen region [Figure 1]. central giant cell granulomas therefore the decision for
surgical excision was taken. The operation was performed
A posterior-anterior radiography of the skull was obtained under general anaesthesia. A preauricular incision was
revealing a radiolucent region in the condyle of the right performed to identify the ATM joint capsule and thereaf-
mandible [Figure 2]. A three-dimensional CT reconstruc- ter the condyle. This structure appeared deformed and
tion showed the presence of a radiolucent and multilocu- increased. The mandibular cortex overlying the cyst was
lar lesion in the condyle region. It was possible to noted to be thin and actually translucent in some places.

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Figure
Three-dimensional
of
thea coronoid
radiolucent
4 andand
part
CT
multilocular
of
reconstruction
the ramus
lesion showing
in the condyle
the presence
region,
Three-dimensional CT reconstruction showing the
Figure 2
Posterior-anterior radiography of the skull presence of a radiolucent and multilocular lesion in
Posterior-anterior radiography of the skull. Radiolu- the condyle region, the coronoid and part of the
cent lesion of the right condyle. ramus. Note the honeycomb appearance and septae within
the bony lesion.

Upon removal of the outer cortex of bone, thick greenish

fluid was encountered, immediately followed by brisk Patient's occlusion did not change after surgery however it
hemorrhage which was difficult to control. Afterwards a was evident that the mandible shifted on the right side
low condylectomy was performed. The lesion underwent during opening [Figure 9]. The patient underwent throw
a complete surgical excision [Figures 7, 8]. The final histo- physiotherapy and functional treatments with Bionator.
logical diagnosis was aneurysmal bone cyst described as This device helped the boy to have a good occlusion dur-
an expanding osteolytic lesion containing blood-filled ing his growth. The patient came monthly to our ortho-
spaces of variable size, separated by connective tissue con- dontic department for functional exercises and to upgrade
stituted by bone trabeculae or osteoid tissue and many the Bionator.
osteoclastic giant cells.
Discussion
Although the aneurysmal bone cyst is a lesion relatively
common in the skeletal structure, is not usual to find it in
the facial region of the skeleton. In 1893, Van Arsdale [15]
called this lesion "homerus ossifying haematoma". In

Figure
Three-dimensional
of
thea coronoid
radiolucent
3 andand
part
CT
multilocular
of
reconstruction
the ramus
lesion showing
in the condyle
the presence
region, Figure
Three-dimensional
of
thea coronoid
radiolucent
5 andand
part
CT
multilocular
of
reconstruction
the ramus
lesion showing
in the condyle
the presence
region,
Three-dimensional CT reconstruction showing the Three-dimensional CT reconstruction showing the
presence of a radiolucent and multilocular lesion in presence of a radiolucent and multilocular lesion in
the condyle region, the coronoid and part of the the condyle region, the coronoid and part of the
ramus. Note the honeycomb appearance and septae within ramus. Note the honeycomb appearance and septae within
the bony lesion. the bony lesion.

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periphery
Magnetic
sity
Figure
within
6 resonant
the lesion
imaging
itself, (MRI)
with a showing
low-signal
a high-signal
intensity atinten-
the
Magnetic resonant imaging (MRI) showing a high-sig-
nal intensity within the lesion itself, with a low-signal Figure
Intraoperative
cal plate7overlying
exposure
the mandibular
of the thin,
cyst
translucent lateral corti-
intensity at the periphery. Intraoperative exposure of the thin, translucent lat-
eral cortical plate overlying the mandibular cyst.
Afterwards a low condylectomy, a coronoid process and
1940 Ewing used the term "aneurismal" to describe such mandibular ramus curettage were performed.
lesion [16]. In 1942, Jaffé and Lichtenstein [13] used the
term "aneurismal cyst" and in 1950 they coined the term
"aneurismal bone cyst" for the first time in literature.
There are presented about 160 cases of aneurysmal bone sarcoma, globulomaxillary cyst, hamangioendothelioma,
cyst in the maxillofacial region in world literature [[1,3] and hemangiopericytoma [2,10]. The initial diagnosis can
and [4]]. be made radiographically, with MRI being considered as
the first choice diagnostic too. However definitive diagno-
Clinical presentations of the ABC range from mild, slowly sis requires histopathological examination of the surgical
expanding, semisolid growth causing slight facial asym- specimen.
metry to a rapidly expanding vascular swelling causing
extensive bone destruction and mimicking malignant It is important to note that, to make the diagnosis of aneu-
lesions [10]. This lesion does not have a clear clinical spe- rysmal bone cyst, it is mandatory to take into account
cificity. The radiographic appearance of the vascular ABC patient's history, physical examination, radiographic and
typically demonstrates an expansive lesion with thinning histopathologic evaluation [19].
of cortical plate and a honeycomb or soap bubble appear-
ance [17,18]. The growth of the upper jaw, after surgery, was regular and
seemed to not be influenced by the shifting movement of
These characteristics like the sudden growth, cortical the mandible during opening. The forward translation of
destruction, osteoid formation, and tumour-like appear- the healthy left condyle during opening operated by the
ance, can easily cause confusion with malignancy. left lateral pterygoid muscle was responsible for a man-
dibular shifting to the side were it was performed the con-
It is important to differentiate the ABC from other pathol- dylectomy.
ogies that occur in the maxillofacial region. These include
peripheral and central giant cell reparative granuloma, At present the patient is under control for any recurrence
traumatic bone cyst, brown tumor of hyperparathy- of the lesion. The differential diagnosis of aneurysmal
roidism, myxoma, fibrous dysplasia, desmoplastic bone cyst from malignant tumours is the main practical
fibroma, fibrous histiocytoma, hemangioma, osteogenic aspect of this osteolytic lesion.

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Figure
Intraoperative
cal plate8overlying
exposure
the mandibular
of the thin,
cyst
translucent lateral corti-
Intraoperative exposure of the thin, translucent lat-
eral cortical plate overlying the mandibular cyst.
Afterwards a low condylectomy, a coronoid process and
mandibular ramus curettage were performed.

Moreover the functional device Bionator is expected to

stimulate the operated right side of the mandible for an
additional growing. There is a vast documentation how
functional appliances are used for hemifacial microsomia
with excellent results [20]. The condyle and the ramus of
the mandible have great potential of growing if oppor-
tunely stimulated [21].
Figureopening
Post-operative
during 9 image
towards
showing
the operated
the shifting
side of the mandible
Post-operative image showing the shifting of the
A reconstruction of the right condyle will be taken in con-
mandible during opening towards the operated side.
sideration as last option by the end of patient's growth
only if the functional therapy will fail.

The authors warn that massive or excessive hemorrhage

may complicate surgery and be difficult to control [5,10]. recurrence rate within literature [1-23]. This article sug-
Similar intra-surgery complication have been described in gests how a complete surgical resection can definitively
others publications. Good surgical exposure and control eradicate this aggressive bone lesion with a quite high
of any feeding vessel that can be identified close to the recurrence rate. In conclusion surgical excision is the treat-
lesion facilitate excision, minimize blood loss, and aid in ment of choice and the differential diagnosis of ABC from
the preservation of vital structures. This case demon- malignant tumours is the most important clinical aspect.
strated the aggressive and destructive behaviour common No recurrence was observed at a 3 years follow-up.
to vascular ABC of the jaw.
Consent
The treatment of this lesion consist of complete surgical Written informed consent was obtained from the patient
excision, it demonstrates a low recurrence rate. It has been and patient's parents for publication of this case report
proposed radiation therapy however the risk of subse- and accompanying images. A copy of the written consent
quent malignant degeneration is present. It has been is available for review by the Editor-in-Chief of this Jour-
reported sarcoma arisen within radiated ABC [[1,3] and nal.
[4]]. Even curettage of the cysts has a recurrence rate as
high as 50%. Technical difficulties in entirely removing Competing interests
very large lesions can be the explanation for very different The authors declare that they have no competing interests.

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Authors' contributions
SP, GG and PFA performed the surgery and carried out the
case study. PFA and RB wrote the article. AM reviewed sci-
entific literature for this mandibular cyst. All the authors
have read and approved the manuscript.

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