YIJOM-1526; No of Pages 5

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

doi:10.1016/j.ijom.2009.01.014, available online at http://www.sciencedirect.com

Case Report
Clinical Pathology

Recurrent aneurysmal bone T. Ettl1, K. Ständer1, S. Schwarz2,

T. E. Reichert1, O. Driemel1
1
Department of Oral and Maxillofacial

cyst of the mandibular condyle

Surgery, Regensburg University, Germany;
2
Department of Pathology, Erlangen
University, Germany

with soft tissue extension

T. Ettl, K. Ständer, S. Schwarz, T. E. Reichert, O. Driemel: Recurrent aneurysmal bone
cyst of the mandibular condyle with soft tissue extension. 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 case of a 17- year-old female patient with a destructive aneurysmal
bone cyst of the right mandibular condyle is presented. Surgery revealed an eggshell
thin, partly perforated remaining condylar cortex with extension of the lesion into
the surrounding soft tissues. Condylar resection with curettage of the adjacent soft
tissues and recurrence prophylaxis with intranasal calcitonin for 1 week could not
prevent recurrence after 6 months. In a second operation the ascending mandibular Keywords: aneurysmal bone cyst; condyle;
ramus was partly resected and immediately reconstructed with a newly developed recurrence; calcitonin; condylar head add-on.
alloplastic condylar head add-on system. This time the lesion was intraoperatively
completely surrounded by solid cortical bone. Accepted for publication 29 January 2009

An aneurysmal bone cyst (ABC) is an
expansile, often multilocular, osteolytic
lesion, with blood-filled spaces separated
by fibrous septa containing giant cells
and reactive bone9. Its occurrence in
the maxillofacial region is uncommon,
with fewer than 100 reported cases in the
jaw and 9 originating in the condyle. The
aetiology and pathogenesis are elusive.
The lesion can cause large lytic bony
defects and tends to recur after surgery.
This case presents a rare ABC of the right
mandibular condyle, the application of
intranasal calcitonin for recurrence pro-
phylaxis and immediate condylar recon-
struction with a new alloplastic condylar
head add-on system5.
Case report
A 17-year-old female patient was referred
with a 2-month history of painful swelling
in the right temporomandibular joint
(TMJ) region and a radiolucent lesion in
the area of the right mandibular condyle
on a panoramic radiographic examination
(Fig. 1a and b). Clinical examination
revealed a smooth 2.5  3.5 cm swelling
in the right preauricular region with ten-
derness and cracking on palpation. The
patient showed limited interincisal mouth
opening (25 mm), bilateral non-occlusion
and a mandibular shift to the left side
(Table 1). Cranial nerves V and VII were
intact. Submandibular and cervical lymph
nodes were not palpable. The patient did
not report any history of weight loss,
infection or iatrogenic dental trauma.
Blood tests were unremarkable. Computed
tomography (CT) and magnetic resonance
imaging (MRI) revealed an expanded,
cystic 4.3  3.9  3.6 cm lesion of the
right mandibular condyle with septa and
fluid levels, suggesting an ABC (Fig. 1c).
A punch biopsy was performed and
histopathological examination showed a
benign, fibrous tumour containing giant
cells and reactive bone (Fig. 2a). After
additional immunohistochemical exami-
nation (S100 negative, MIB-1 about
10%) an osteosarcoma was excluded
and in the light of the radiographic and

0901-5027/000001+05 $36.00/0 # 2009 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: T.. Ettl, et al., Recurrent aneurysmal bone cyst of the mandibular condyle with soft tissue extension,
Int J Oral Maxillofac Surg (2009),
YIJOM-1526; No of Pages 5

2 Ettl et al.

Fig. 1. (a) Right preauricular swelling. (b) Panoramic radiograph showing a large unilocular radiolucency of the right condyle with a thinned
corticalis. (c) MRI with contrast medium shows the lesion with septa and fluid level.

immunohistochemical findings an ABC
was diagnosed.
Under general nasotracheal anaesthesia,
the lesion was approached via an extraoral
preauricular and mandibular incision.
Intraoperatively the remaining condylar
cortex was eggshell thin, partly perforated
and could hardly be separated from the
surrounding soft tissues (Fig. 2b). The
condylar processus was resected and the
adjacent soft tissue curetted (Fig. 2c). The
articular disc was not invaded and was left
intact in the glenoid fossa. No primary
reconstruction of the condyle was per-
formed. In accordance with the manufac-
turer’s recommendation, a twice daily
application of salmon calcitonin nasal
spray (Miacalcin1, Novartis Pharma,
100 IU, for 1 week) was started for recur-
rence prophylaxis.
After a follow up of 6 months and the
absence of clinical symptoms, panoramic
radiographic examination and MRI
revealed a large recurrence, 4.5 cm in

Table 1. Condylar head add-on documentation - functional data.

Post-Op Post-Op Post-Op
Pre-Op (1st Op) 2 weeks (2nd Op) 2 weeks (2nd Op) 8 months
Max. Interincisal distance (mm) 25 32 23 34
Midline deviation at habitual occlusion (mm) 1 (left) 2 (right) 0 1 (right)
Midline deviation at Max. Interincisal distance (mm) 2 (right) 9 (right) 5 (right) 6 (right)
Max. lateral movement to the right (mm) 11 11 11 11
Max. lateral movement to the left (mm) 6 3 3 4
Max. protrusion (mm) 3 2 4 4
Overjet/Overbite (mm) 0/0 2/2 2/2 2/2

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Int J Oral Maxillofac Surg (2009),
YIJOM-1526; No of Pages 5
Fig. 2. (a) Microphotograph of the histologic specimen. Vascular space without epithelial
lining, partly filled with red blood cells. Surrounded by fibroblast-rich connective tissue with
osteoid and multinuclear giant cells (hematoxylin and eosin stain, magnification 200). (b)
Intraoperative site showing the preauricular lesion caudal to the zygomatic process. (c) Irregular
resection specimen without visible bony cortex.
diameter (Fig. 3a). In further surgery the
right ascending mandibular ramus was
partly resected and the condyle was recon-
structed by a condylar head add-on system
(Synthes GmbH, Umkirch, Germany).
Intraoperatively, in contrast to the first
surgery, the lesion was completely sur-
rounded by solid cortical bone and could
easily be separated from the surrounding
soft tissues (Fig. 3b).
In the last follow up, 8 months after the
second operation, the patient was free of
recurrence (Fig. 3c) with the functional
data listed in Table 1.
Discussion
The incidence of the solid type of ABC has
been reported as 5–8%22, usually occur-
ring in young patients below the age of 20
years. In the maxillofacial skeleton, most
lesions occur in the mandible, predomi-
nantly in the posterior region including the
ascending ramus22. The condyle is rarely
affected6,15. The aetiology of ABCs
Please cite this article in press as: T.. Ettl, et al., Recurrent aneurysmal bone cyst of the mandibular condyle with soft tissue extension,
Int J Oral Maxillofac Surg (2009),
Recurrent aneurysmal bone cyst of the mandibular condyle
remains elusive. Primary origin,15 as well
as secondary development, in a pre-exist-
ing bone lesion (e.g. giant cell lesion)13
occurs. Haemodynamic disorders and
arteriovenous malformations are thought
to increase intraosseous venous pressure
with expansion of the vascular bed, lead-
ing to bone resorption and replacement by
connective tissue and reactive osteoid14.
In recent years, chromosomal alterations
of segments 17p and 16q have been
described suggesting a neoplastic origin
of the lesion1.
Clinically ABC presents as a firm swel-
ling with or without tenderness, progres-
sively enlarging and occasionally
perforating the bony cortex. Malocclusion
and tooth displacement are common. The
radiographic appearance of mandibular
ABC is variable. Unicystic or multicystic,
mostly radiolucent lesions with thinning
and destruction of the bony cortices and
with well-defined or diffuse margins have
often been reported10,15. CT may help to
define the exact extent of the lesion’s
3
borders and MRI may reveal the charac-
teristic fluid levels and septa19,22, as in this
case. Radiographic findings are suggestive
but not diagnostic, as there are other enti-
ties with similar radiological appearance.
The main differential diagnoses include
ameloblastoma, myxoma, central giant
cell granuloma, odontogenic cyst and
venous malformation of the bone12.
Diagnosis is established by histopatho-
logical examination of a tissue sample
(fine needle, punch or incisional biopsy).
Microscopically blood-filled cavities are
lined by macrophages and not by endothe-
lial cells. The cavities are separated by
septa, composed of fibroblasts, osteoclast-
like giant cells and reactive bone or irre-
gular osteoid9.
Surgery is the treatment of choice. The
extent of the operation depends on the site
and size of the cyst. In smaller lesions and
those confined in bone conservative cur-
ettage may be preferred. Radiation therapy
should not be used owing to the increased
risk of developing radiation-induced sar-
comas19. For surgically inaccessible
lesions percutaneous embolization has
been used with encouraging results7.
Recurrence rates for ABC are relatively
high and exceed 30%19. Most recurrences
develop within 6–9 months, especially if
the lesion shows perforated cortices and
extends into adjacent soft tissue, which
aggravates its eradication15. Owing to the
low incidence of ABC and lack of com-
parative studies, little is known about
recurrence rates following different treat-
ment regimes.
In the present case, intranasal salmon
calcitonin was used for recurrence prophy-
laxis. Calcitonin inhibits osteoclastic
activity and promotes trabecular bone for-
mation with increased mineral density2. It
is mainly used for postmenopausal osteo-
porosis and given by subcutaneous and
intralesional injection or intranasal spray.
In ABC and giant cell lesions good results
are reported with calcitonin injec-
tions8,17,18,20. POGREL et al.17 observed
complete resolution of mandibular central
giant cell granuloma following daily sub-
cutaneous calcitonin injections (100 IU)
after a 20-month treatment period in 8 of 9
cases. The successful application of intra-
lesional calcitonin, with complete ossifi-
cation after 6 months, is described for
vertebral ABC18. In contrast, a recurrent
ABC of the TMJ region showed no
response to a 9-month application of sub-
cutaneous calcitonin injections19. Intrana-
sal calcitonin is associated with less
frequent side-effects (e.g. flushing, tin-
gling, nausea) than parenteral application
and is increasingly being used. DOMINGUEZ
YIJOM-1526; No of Pages 5

4 Ettl et al.

Fig. 3. (a) Panoramic radiograph 6 months after first surgery. Soap bubble-like lesion within expanded condyle and visible cortex. (b) Surgical
resection specimen with septed spaces and solid bony surrounding. (c) Panoramic radiograph 8 months after second operation. Alloplastic
reconstruction of the condyle by a condylar head add-on system.

et al.4 reported complete resolution in
three cases of maxillofacial giant cell
lesions after 12–19 months’ 100 IU intra-
nasal calcitonin, however, in two recent
studies, no response to intranasal applica-
tion was observed for this lesion after 13–
64 months3,21.
In the present case, calcitonin nasal
spray was used for only 1 week, which
could not prevent recurrence, but the
lesion was surrounded by solid cortical
bone at the second operation after 6
months. It is not known whether this bony
regeneration was related to the calcitonin
and if longer application, higher dose or
use of subcutaneous or intralesional injec-
tions might have resulted in complete
remission. Spontaneous ossification of
ABC is described in the literature16 and
may explain this cortical regeneration.
Surgical treatment of the recurrent man-
dibular ABC should be more radical. In
many cases partial mandibular resection
and reconstruction is required. For recon-
struction of the mandibular ascending
ramus and the condyle, the use of costo-
chondral rib grafts or vascularized fibula
free-flaps with Alloderm neocondyle
reconstructions has had favourable
results6,11,19. This case documents the first
application of an alloplastic condylar head
add-on system for immediate, temporary
reconstruction in ABC. The height-adjus-
table positioning of the condylar head add-
on and its offset in a medial direction
allows anatomically correct positioning5.
No secondary donor site is needed,
decreasing surgery time and physical ther-
apy can begin immediately. The system
provides functional and aesthetic condylar
rehabilitation until recurrence can be
excluded and a definite autogeneous
reconstruction can be performed.
In conclusion, this case presents a rare
aneurysmal bone cyst of the mandibular
condyle. Extension of the lesion into
adjacent soft tissue increases the risk of
recurrence. Calcitonin may lead to cortical

Please cite this article in press as: T.. Ettl, et al., Recurrent aneurysmal bone cyst of the mandibular condyle with soft tissue extension,
Int J Oral Maxillofac Surg (2009),
YIJOM-1526; No of Pages 5
re-ossification and simplify surgery. Tem-
porary reconstruction of the condyle with
the newly developed alloplastic condylar
head add-on system provides functional
and aesthetic rehabilitation of the TMJ
system.
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Address:
Tobias Ettl
Klinik und Poliklinik für Mund-
Kiefer- und Gesichtschirurgie
Klinikum der Universität Regensburg
Franz-Josef-Strauß-Allee 11
93053 Regensburg
Germany
Tel: +49 0941 9446329
Fax: +49 0941 9446302
E-mail: et200@gmx.de