J Clin Exp Dent. 2019;11(6):e561-4.

Aneurysmal bone cyst of the mandible with conservative surgical management

Journal section: Oral Medicine and Pathology doi:10.4317/jced.55771

Publication Types: Case Report http://dx.doi.org/10.4317/jced.55771

Aneurysmal bone cyst of the mandible with conservative

surgical management: A case report

Efraín-del Cristo Álvarez-Martínez 1, Mónica-Vanessa Posso-Zapata 2, Vanessa-Andrea Flórez-Arango 2,

Johan-Sebastián Lopera-Valle 3, Carlos-Martín Ardila 4

1
Oral and maxillofacial surgeon. University of Antioquia. Medellín, Colombia
2
Postgraduate student of Oral and Maxillofacial Surgery. University of Antioquia. Medellín, Colombia
3
Postgraduate student of Radiology. University of Antioquia. Medellín, Colombia
4
Periodontist, Ph.D. in Epidemiology, Coordinator of the Biomedical Stomatology Research Group. Medellín, Colombia

Correspondence:
Calle 70 No. 52-21, Medellín, Colombia
[email protected]
Álvarez-Martínez E, Posso-Zapata MV, Flórez-Arango VA, Lopera-Valle
JS, Ardila CM. Aneurysmal bone cyst of the mandible with conservative
Received: 03/04/2019 surgical management: A case report. J Clin Exp Dent. 2019;11(6):e561-4.
Accepted: 02/05/2019 http://www.medicinaoral.com/odo/volumenes/v11i6/jcedv11i6p561.pdf

Article Number: 55771 http://www.medicinaoral.com/odo/indice.htm

© Medicina Oral S. L. C.I.F. B 96689336 - eISSN: 1989-5488
eMail: [email protected]
Indexed in:
Pubmed
Pubmed Central® (PMC)
Scopus
DOI® System

Abstract
The aneurysmal bone cyst is a nonneoplastic, osteolytic and locally destructive lesion that mainly affects the me-
taphyseal area of long bones and only 2% of it is diagnosed in the maxillofacial skeleton. Although surgical treat-
ment is the most common option, it is associated to high morbidity rates. The case of an aneurysmal bone cyst of
a considerable size in a 27-year-old male patient illustrating a conservative surgical approach with preservation of
the dental structures in the mandible to limit aesthetic and functional side effects is presented. Two-year clinical
follow-up was performed with no evidence of recurrence.

Key words: Aneurysmal bone cyst; curettage, conservative treatment, mandibular osteotomy.

Introduction the mandible (2,3). According to the World Health Or-

The aneurysmal bone cyst (ABC) is a benign intraos- ganization, this lesion is classified within the giant-cell
seous lesion that develops in patients under 30 years, its lesions, together with central and peripheral giant-cell
location is usually the metaphysis of long bones and only granuloma, cherubism and simple bone cyst (4).
2% of cases are diagnosed in the maxillofacial skeleton; ABC is a rare, expansive, locally destructive lesion and
it represents 1,5% of nonodontogenic maxillary cysts, it constitutes a diagnostic and therapeutic challenge in
and has a 3:1 mandible/maxilla rate (especially in the the daily clinical practice (2). Therapeutic management
posterior area) (1). for ABC is still controversial; the approach depends on
The ABC is generally diagnosed in young people be- factors such as age, location, extent and size of the le-
tween their twenties and thirties, has no predilection for sion. Several treatment options, such as simple curetta-
any gender, usually affects long bones (50%), vertebral ge, en bloc resection, radiation therapy, embolization or
column (20%), and around 2% manifests in the bones of a combination of these methods, have been proposed.

e561
J Clin Exp Dent. 2019;11(6):e561-4. Aneurysmal bone cyst of the mandible with conservative surgical management

Although surgical treatment is the most common alter- Panoramic radiography revealed a multilocular radiolu-
native, it is associated to high morbidity rates including cent lesion with no defined borders, extending from the
postoperative aesthetic and functional alterations, usua- mesial root of tooth #36 to tooth #44, that affected the
lly as a consequence of extensive resections, in expansi- interradicular bone, with root resorption of teeth #31,
ve lesions. This article presents the case of a mandibular #41 and #42 (Fig. 1b).
ABC of significant size successfully managed through With a diagnostic impression of ameloblastoma versus
a conservative surgical approach aimed at limiting the central giant-cell granuloma, a biopsy was taken under
aesthetic and functional side effects in a young patient. local anesthesia after 4cc aspiration of dark hematic se-
cretion. Histopathological analysis confirmed the ABC
Case Report diagnostic; blood-filled vesicular channels surrounded
27-year-old male patient with no underlying systemic disea- by lamellar bone with spicules surrounded in turn by
ses who sought medical attention because of a three-month osteoblasts were described. Multinucleated giant cells
medical case of facial asymmetry from increased volume and abundant fibroblasts were also observed (Fig. 1c).
of the left chin area (Fig. 1a), with no other associated Additionally, the CT scan evidenced this expansive lytic
symptoms. The patient claimed he had been subject to fa- lesion of 29 x 45 x 30 mm associated to a perforation of
cial trauma in the same area two years before. the vestibular and lingual bone tables (Fig. 1d).
Adequate mouth opening and mandibular mobility were Considering the patient’s age, the dimensions of the
found in the clinical examination; no reactive lympha- lesion and the potential functional and aesthetic con-
denopathies were found. Permanent dentition, stable oc- sequences of the en bloc resection, bone curettage and
clusion and displacement and rotation of tooth #33 were additional endodontic treatment of the anteroinferior
observed in the intraoral examination; swelling of the teeth was performed, anticipating the compromise of the
soft tissues and effacement of the bottom of the sulcus vascular and nervous contribution due to the extension
between teeth #34 and #42 were found. The oral mucosa of the lesion to the apex of these teeth; Likewise, a se-
was found to be healthy, smooth, firm and with no pain mi-rigid wire and resin splint was used to stabilize the
during palpation. dentoalveolar segment.

Fig. 1: a. Pre-surgical evaluation with evident asymmetry of the lower third, mainly the left side. b. Pre-
treatment panoramic radiograph with multilocular radiolucent lesion showing irregular edges and extend-
ing from the mesial root of tooth #36 to #44 with compromised interradicular bone. Note the root resorption
of teeth #31, #41 and #42. c. Histopathology study (Hematoxylin Eosin) with blood-filled vesicular channels
surrounded by lamellar bone, with spicules surrounded in turn by osteoblasts. Multinucleated giant cells and
abundant fibroblasts are also described. d. 3D reconstruction of front-view computed tomography showing
29 x 45 x 30 mm expansive lithic lesion related to the perforation of the vestibular and lingual bone tables.

e562
J Clin Exp Dent. 2019;11(6):e561-4. Aneurysmal bone cyst of the mandible with conservative surgical management

A circumvestibular surgical incision of approximately le maintaining a stable occlusion, feeding was achieved
34 to 43 was performed under general anesthesia to gain via nasogastric tube for 15 days.
access to the lesion; a highly vascularized lesion with After two months of postoperative follow-up, an 8mm
evident angiogenesis was observed; profuse bleeding dehiscence with root exposure of tooth #33 was obser-
was controlled through local procedures and cauteriza- ved in the vestibular area, so it was extracted considering
tion. Once the tumor was removed, the surgical site, in- its poor prognosis. The patient was evaluated through to-
cluding the basilar rim, was extended 5mm; to stabilize mographies (Fig. 3a,b), clinically (Fig. 3c) and radiogra-
the mandible, two 2.7 mm preformed reconstruction pla- phically (Fig. 3d) for two years, no tumoral recurrence
tes associated to the filling of the defect with bone graft was identified during that time; aesthetic and functional
were placed. Flaps were repositioned and the procedure results were satisfactory. A written consent of the patient
finished with no immediate complications (Fig. 2). Whi- according to ethical principles was signed.

Fig. 2: a. Aneurysmal bone cyst removal. b Surgical site with preservation of dental structures.

Fig. 3: a,b. 3D reconstruction of front-view computed tomography 6 months after the

postoperative check-up showing adequately positioned titanium plates and ongoing bone
remodeling. c. Twenty-four months of postoperative follow-up, with no facial asymmetry.
d. Panoramic radiograph after 24 months of postoperative follow-up showing bone defect
in the surgical site associated with corticalization of the edges and absence of residual
lesions.

e563
J Clin Exp Dent. 2019;11(6):e561-4. Aneurysmal bone cyst of the mandible with conservative surgical management

Discussion tence of residual lesion, considering the absence of we-

The etiology and pathogenesis of ABC is not very clear. ll-defined edges or capsule at the time of the procedure
The most accepted theory is the existence of previous (11,12). In this case, no recurrence was observed during
trauma (5,6), which corresponds to what the patient re- the follow-up of two years, which is the time in which
ported in this case. Likewise, as described in the litera- most scientific evidence reports it (11-15).
ture, it also develops in young patients (2,3).
In this case, correspondence with the literature was also References
observed, as the ABC usually develops as an expansive, 1. Sun ZJ, Sun HL, Yang RL, Zwahlen RA, Zhao YF. Aneurysmal
bone cysts of the jaws. Int J Surg Pathol. 2009;17:311-22.
well-defined, uni or multilocular lytic lesion with thin 2. Flores IL, Hamilton ME, Zanchin-Baldissera E, Uchoa-Vasconcelos
sclerotic margins, liquid-liquid levels, as well as diffe- AC, Chaves-Tarquinio SB, Neutzling-Gomes AP. Simple and aneurys-
rent degrees of cortical rupture (Fig. 1d) and extension mal bone cyst: Aspects of jaw pseudocysts based on an experience of
to adjacent soft tissues (6). Brazilian pathology service during 53 years. Med Oral Patol Oral Cir
Bucal. 2017;22:e64-e9.
Histological characteristics are related to the replace- 3. Bharadwaj G, Singh N, Gupta A, Sajjan AK. Giant aneurysmal bone
ment of bone with fibro-osseous tissue and multinuclea- cyst of the mandible: A case report and review of literature. Natl J
ted giant cells, with blood-filled sinusoidal or cavernous Maxillofac Surg. 2013;4:107-10.
spaces (2,3,4,7), as observed in this case (Fig. 1c). 4. El-Naggar AK, Chan JKC, Takata T, Grandis JR, Slootweg PJ. The
fourth edition of the head and neck World Health Organization blue
Considering the differential diagnosis, it is important to book: editors’ perspectives. Hum Pathol. 2017;66:10-2.
bear in mind that ABCs usually expand to a greater de- 5. Marín Fernández AB, García Medina B, Martínez Plaza A, Agui-
gree and are more frequent in the posterior aspect of the lar-Salvatierra A, Gómez-Moreno G. Aneurysmal bone cyst of the
mandible than giant-cell granulomas, while ameloblas- mandible affecting the articular condyle: a case report. Clin Case Rep.
2016;4:1175-80.
toma is more frequent in older patients, and cherubism 6. Aadithya U, Jeyaseelan A, Himanshi C. Aneurysmal Bone Cyst
is a bilateral multifocal disease (8). of the Jaws: Clinicopathological Study. J Maxillofac Oral Surg.
The treatment for ABC depends on the age, extension, 2014;13:458-463
aggressiveness, size and location of the lesions; It ran- 7. Grecchi F, Zollino I, Candotto V, Gallo F, Rubino G, Bianco R.
A case report of haemorrhagic-aneurismal bone cyst of the mandible.
ges from simple curettage to extensive resection with Dent Res J (Isfahan). 2012;9:S222-4.
subsequent rehabilitation (9-12). Most ABCs are asso- 8. An SY. Aneurysmal bone cyst of the mandible managed by conser-
ciated with another pathological entity such as ossifying vative surgical therapy with preoperative embolization. Imaging Sci
fibroma, central giant-cell granuloma or benign osteo- Dent. 2012;42:35-9.
9. Costa de Freitas RM, Fonseca KC, Procópio RJ, Cardoso Lehman
blastoma, which conditions the aggressiveness of the LF. Image-Guided Injection of Bone Allograft and Autologous Bone
treatment to be considered (1). Cottalorda and Bourelle Marrow for the Treatment of Aneurysmal Bone Cyst of the Jaw. J Vasc
(13) suggested that inactive lesions can only be cured Interv Radiol. 2017;28:1299-302.
with biopsy or curettage; however, in active or aggres- 10. Park HY, Yang SK, Sheppard WL, Hegde V, Zoller SD, Nelson
SD, et al. Current management of aneurysmal bone cysts. Curr Rev
sive lesions, resection offers a satisfactory theoretical Musculoskelet Med. 2016;9:435-44.
solution (14). 11. Grecchi E, Borgonovo A, Re D, Creminelli L. Aneurismal bone
One of the therapeutic challenges of the ABC aimed cyst: a conservative surgical technique. A case report treated with a
at reducing postoperative morbidity involves avoiding small access osteotomy. Eur J Paediatr Dent. 2016;17:100-3.
12. Docquier PL, Delloye C. Treatment of aneurysmal bone cysts by
damage to the alveolar neurovascular bundle included introduction of demineralized bone and autogenous bone marrow. J
or adjacent to the neoplastic lesion. Similarly, rigorous Bone Joint Surg Am. 2005;87:2253-8.
resection or curettage in all cyst walls should be oriented 13. Cottalorda J, Bourelle S. Current treatments of primary aneurysmal
towards preserving as many dental pieces as possible, bone cysts. J Pediatr Orthop B. 2006;15:155-67.
14. Liu K, Guo C, Guo R, Meng J. A Giant Aneurysmal Bone Cyst in
without considerably increasing the risk of recurrence the Mandibular Condyle. J Craniofac Surg. 2017;28:e148-e51.
(15). 15. Rațiu C, Ilea A, Gal FA, Ruxanda F, Boşca BA, Miclăuș V. Man-
In this case, considering the patient’s age, the compro- dibular aneurysmal bone cyst in an elderly patient: Case report. Gero-
mised area and future oral and facial rehabilitation, a dontology. 2018;35:143-6.
conservative surgical management was chosen. The Conflict of interest
treatment with bone curettage, in combination with the The authors have declared that no conflict of interest exist.
performed endodontics had a positive response, the le-
sion healed and an adequate facial aesthetic with preser-
vation of masticatory function and phonation was achie-
ved. In this case, an en bloc resection with anteroinferior
dental loss that would have required more complex ins-
tances of rehabilitation due to the high degree of aesthe-
tic and functional consequences was avoided.
The highest ABC recurrence rates (21%) (10) have been
reported in relation to bone curettage due to the persis-

e564