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

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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 surrounding soft tissues (Fig. 2b). The application of salmon calcitonin nasal
was diagnosed. condylar processus was resected and the spray (Miacalcin1, Novartis Pharma,
Under general nasotracheal anaesthesia, adjacent soft tissue curetted (Fig. 2c). The 100 IU, for 1 week) was started for recur-
the lesion was approached via an extraoral articular disc was not invaded and was left rence prophylaxis.
preauricular and mandibular incision. intact in the glenoid fossa. No primary After a follow up of 6 months and the
Intraoperatively the remaining condylar reconstruction of the condyle was per- absence of clinical symptoms, panoramic
cortex was eggshell thin, partly perforated formed. In accordance with the manufac- radiographic examination and MRI
and could hardly be separated from the turer’s recommendation, a twice daily 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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Recurrent aneurysmal bone cyst of the mandibular condyle 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
Fig. 2. (a) Microphotograph of the histologic specimen. Vascular space without epithelial
aggravates its eradication15. Owing to the
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) low incidence of ABC and lack of com-
Intraoperative site showing the preauricular lesion caudal to the zygomatic process. (c) Irregular parative studies, little is known about
resection specimen without visible bony cortex. recurrence rates following different treat-
ment regimes.
In the present case, intranasal salmon
diameter (Fig. 3a). In further surgery the remains elusive. Primary origin,15 as well calcitonin was used for recurrence prophy-
right ascending mandibular ramus was as secondary development, in a pre-exist- laxis. Calcitonin inhibits osteoclastic
partly resected and the condyle was recon- ing bone lesion (e.g. giant cell lesion)13 activity and promotes trabecular bone for-
structed by a condylar head add-on system occurs. Haemodynamic disorders and mation with increased mineral density2. It
(Synthes GmbH, Umkirch, Germany). arteriovenous malformations are thought is mainly used for postmenopausal osteo-
Intraoperatively, in contrast to the first to increase intraosseous venous pressure porosis and given by subcutaneous and
surgery, the lesion was completely sur- with expansion of the vascular bed, lead- intralesional injection or intranasal spray.
rounded by solid cortical bone and could ing to bone resorption and replacement by In ABC and giant cell lesions good results
easily be separated from the surrounding connective tissue and reactive osteoid14. are reported with calcitonin injec-
soft tissues (Fig. 3b). In recent years, chromosomal alterations tions8,17,18,20. POGREL et al.17 observed
In the last follow up, 8 months after the of segments 17p and 16q have been complete resolution of mandibular central
second operation, the patient was free of described suggesting a neoplastic origin giant cell granuloma following daily sub-
recurrence (Fig. 3c) with the functional of the lesion1. cutaneous calcitonin injections (100 IU)
data listed in Table 1. Clinically ABC presents as a firm swel- after a 20-month treatment period in 8 of 9
ling with or without tenderness, progres- cases. The successful application of intra-
sively enlarging and occasionally lesional calcitonin, with complete ossifi-
Discussion
perforating the bony cortex. Malocclusion cation after 6 months, is described for
The incidence of the solid type of ABC has and tooth displacement are common. The vertebral ABC18. In contrast, a recurrent
been reported as 5–8%22, usually occur- radiographic appearance of mandibular ABC of the TMJ region showed no
ring in young patients below the age of 20 ABC is variable. Unicystic or multicystic, response to a 9-month application of sub-
years. In the maxillofacial skeleton, most mostly radiolucent lesions with thinning cutaneous calcitonin injections19. Intrana-
lesions occur in the mandible, predomi- and destruction of the bony cortices and sal calcitonin is associated with less
nantly in the posterior region including the with well-defined or diffuse margins have frequent side-effects (e.g. flushing, tin-
ascending ramus22. The condyle is rarely often been reported10,15. CT may help to gling, nausea) than parenteral application
affected6,15. The aetiology of ABCs define the exact extent of the lesion’s and is increasingly being used. DOMINGUEZ

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

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Int J Oral Maxillofac Surg (2009),
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Recurrent aneurysmal bone cyst of the mandibular condyle 5

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