Journal of Oral and Maxillofacial Radiology

CASE REPORT
Year
: 2014  |  Volume : 2  |  Issue : 2  |  Page : 64--67

Aneurysmal bone cyst of the mandible: Report of a case with a review of the literature


Hasan Onur Simsek1, Derya Yildirim2, Ozlem Gormez2, Esra Yuce3, Fatma Nilgun Kapucuoglu4,  
1 Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, University of Adnan Menderes, Aydin, Turkey
2 Department of Oral and Maxillofacial Radiology, Faculty of Medicine, University of Suleyman Demirel, Isparta, Turkey
3 Department of Oral and Maxillofacial Surgery, Faculty of Medicine, University of Suleyman Demirel, Isparta, Turkey
4 Department of Pathology and Laboratory Medicine, Faculty of Medicine, University of Suleyman Demirel, Isparta, Turkey

Correspondence Address:
Hasan Onur Simsek
Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, University of Adnan Menderes, Aydin
Turkey

Abstract

Aneurysmal bone cyst (ABC), is a rare benign osteolytic bone neoplasm of bone tissue characterized by several sponge-like blood or serum filled, generally non-endothelialized spaces of various diameters that can contain osteoid tissue and osteoclast-like giant cells. In this article, we describe a case of ABC in the mandibular corpus region with clinical, radiological and pathological findings and review the literature concerning the case. A 23-year-old male patient attended to our clinic with the complaint of a mild-pain on the left posterior lower jaw since two months. The superior margin of the lesion was extending between the apical third parts of the roots of the teeth 35-38. Cone beam computed tomography (CBCT) was performed. CBCT Slices revealed an expansile lesion with hydraulic-appearing outer margins above the mandibular canal. Excisional surgical treatment was performed after raising a mucoperiosteal flap and finalized with curette of lesion under local anesthesia. Diagnosis of aneurysmal bone cyst was confirmed by histopathological evaluation. The patient was pain free after six months follow-up, and panoramic radiography showed no evidence of recurrence. The ABC is most common in those regions of the skeleton where there is a relatively high venous pressure and high marrow content. Clinical presentation of the ABC varies from a small, indolent, asymptomatic lesion to rapidly growing, expansile, destructive lesion causing pain, swelling, deformity, neurologic symptoms, pathologic fracture, and perforation of the cortex. As the radiologic and clinical presentation of ABC is extremely variable, histopathologic examination is placed a great emphasis on for the diagnosis.



How to cite this article:
Simsek HO, Yildirim D, Gormez O, Yuce E, Kapucuoglu FN. Aneurysmal bone cyst of the mandible: Report of a case with a review of the literature.J Oral Maxillofac Radiol 2014;2:64-67


How to cite this URL:
Simsek HO, Yildirim D, Gormez O, Yuce E, Kapucuoglu FN. Aneurysmal bone cyst of the mandible: Report of a case with a review of the literature. J Oral Maxillofac Radiol [serial online] 2014 [cited 2020 Jan 20 ];2:64-67
Available from: http://www.joomr.org/text.asp?2014/2/2/64/138653


Full Text

 Introduction



Aneurysmal bone cyst (ABC) is a rare benign osteolytic bone neoplasm of the bone tissue, characterized by several sponge-like blood- or serum-filled, generally non-endothelialized spaces of various diameters that may contain osteoid tissue and osteoclast-like giant cells. [1],[2] The ABCs are infrequent in craniofacial skeleton with regard to other structures like long bones and the spine; only 2% of the ABCs occur in the jaws. The mandible is more frequently affected than the maxilla, with a proportion from 11:9 to 2:1. [3],[4] The body (90% of the cases) and the mandibular ramus (30%) are the main locations, with rare incidences reported in the coronoid process and the mandibular condyle (2%). [7] It represents approximately 1.5% of all non-odontogenic and non-epithelial cysts of the jaws. [5],[6] ABC can be classified into three types. Conventional or vascular type presents as an expansile, rapidly growing destructive lesion causing cortical perforation and soft tissue invasion. The solid type may reveal as a small asymptomatic lesion that is first noticed as radiolucency on routine radiograph or as a small swelling clinically. [7],[8] The third form or mixed variant exhibits features of both the vascular and solid types. It may be a transitory phase of the lesion because sudden activation or rapid enlargement of stable lesions has been reported. [8] In this article, we describe a case of ABC in the mandibular corpus region with its clinical, radiological, and pathological findings. Also, a review of the literature has been presented.

 Case Report



A 23-year-old male patient presented to our clinic with the complaint of a mild pain on the left posterior lower jaw since 2 months. His medical and family history was unremarkable and there was no history of trauma [Figure 1]. There was no evidence of swelling and sensitivity with palpation on extraoral and intraoral examination. Absence of symptoms like fever or suppuration eliminated infection. The patient had a dental examination at a dental hospital a week ago; a panoramic radiograph [Figure 2] was taken and he was directed to our clinic for treatment. Panoramic radiograph of the patient revealed a unilocular, circular radiolucent lesion with well-defined borders on the left mandible. The superior margin of the lesion was extending between the apical third portions of the roots of the teeth 35-38. Cone beam computed tomography (CBCT) (Planmeca ProMax 3D Mid; Planmeca, Helsinki, Finland) was performed. CBCT slices revealed an expansile lesion with a diameter of 27 × 18 × 12 mm. The lesion had a hydraulic-appearing outer margin above the mandibular canal [[Figure 3] a-c]. The panoramic radiograph and CBCT cross-sectional slices demonstrated no displacement or root resorption of the teeth. Electrical pulp test showed that the involved teeth were vital. Fine-needle aspiration biopsy was performed and blood-tinged fluid was obtained, but it was nondiagnostic. Excisional surgical treatment was performed after raising a mucoperiosteal flap and was completed with curettage of lesion under local anesthesia. The flap was repositioned and sutured with silk 3/0. On gross examination, the excised tumor mass, which are pinkish gray and friable, consisted of some sclerotin. Prophylactic antibiotic as well as nonsteroidal anti-inflammatory drug were administered on the following days. The histologic examination of excisional biopsy specimen revealed multiple blood-filled spaces separated by septa of fibrous tissue containing fibroblasts and multinucleated giant cells. Also, numerous small and large vascular spaces lined by endothelial cells and abundant pools of Red Blood Cells (RBC) were seen [Figure 4]. Thereby, diagnosis of ABC was confirmed by histopathologic evaluation. The patient was pain free after 6 months of follow-up, and panoramic radiography [Figure 5] showed no evidence of recurrence.{Figure 1}{Figure 2}{Figure 3}{Figure 4}{Figure 5}

 Discussion



ABC develops mostly in maxillofacial bones depending on high venous pressure and high marrow content. Therefore, it is rarely seen in the skull bones where there is low venous pressure. The mandible is affected three times more when compared to the maxilla. It is frequently observed at the molar and ramus regions of the mandible. [9]

ABC was first described in the literature by Jaffé and Lichtenstein in 1942. [10] The term "aneurysmatic" emphasizes on expansion of the affected bone, which is called the "blow-out effect." The etiology of ABC is controversial. Increased venous pressure and repletion of the vascular bed in the transformed bone caused by the alteration of local hemodynamics was related to resorption, connective tissue replacement, and osteoid formation by Jaffe and Lichenstein. [8],[10] Levy et al. had reported that development of ABC is related to a history of trauma and subperiosteal hematoma formation. Tillman et al. have reported 95 cases with no history of trauma. [8] Also, there was no history of trauma in the present case. Struthers and Shear assumed that ABC was secondary to a pre-existing lesion and that central giant cell granuloma was the most common of these lesions. [6] Swing [11] suggested that ABC is derived from another type of benign lesion like a giant cell tumor in which some changes have made possible the communication between stroma and medullaris vessels. When this communication is maintained, an ABC should be established. If the connection is interrupted, a giant cell granuloma occurs. Panoutsakopoulos et al. have described three cases of ABC with chromosomal anomalies involving band 16q22. [12] Familial incidence of ABC has also been reported in literature. [13],[14]

Clinical presentation of ABC varies from a small, indolent, asymptomatic lesion to rapidly growing, expansile, and destructive lesion causing pain, swelling, deformity, neurologic symptoms, pathologic fracture, and perforation of the cortex. [15] In the present case, mild pain without any infection and intraoral swelling or facial asymmetry was observed. The radiological features of the ABC in the jaws are variable; the expanded bone appears cystic resembling a honeycomb or soap bubble. Destruction or perforation of the cortex and a periosteal reaction can be also seen. [16] It may appear radiolucent, radiopaque, or mixed. In our case, the ABC represented a unilocular radiolucency causing expansion of the cortical plates. Diagnosis based on only radiographic examination may be misleading due to the radiographic appearance being similar to those of other lesions such as ameloblastoma, myxoma, central giant cell granuloma, odontogenic cysts, or central hemangiomas of the bone. [17]

Histologically, the ABC consists of many sinusoidal blood-filled spaces set in a fibrous stroma, with multinucleated giant cells and osteoid. Hemosiderin is present in variable amounts and there is evidence of osteoid and bone formation. The present case is consistent with the "classic or vascular" form described in the literature. [18] Although there are many options concerning the treatment, the gold standard is surgical excision and curettage of the cavity. [19],[20] Motamedi et al. also reported self-healing of cases on long-term follow-up. [15] The usage of radiotherapy is not recommended because of the probability of radioinduced tumors.

Recurrence rates range from 20 to 30% in different groups and it occurs most frequently within the first year after surgery. This is usually associated with insufficient excision or curettage of the lesion, especially in soft tissue invasive cases. Several authors recommend immediate reconstruction of the defect with autogenous grafts in cases of aesthetic deformity and in cases with high risk of fractures and loss of mandibular continuity. [18] Motamedi et al.[15] reported that initial resection is not necessary and have not noted any recurrences following surgical curettage of mandibular lesions. There was no evidence of any residual lesion after 6 months of follow-up. In the present case, treatment of choice was limited to curettage of the mass and the patient was regularly monitored.

 Conclusion



As the radiologic and clinical presentation of ABC is extremely variable, a great emphasis is placed on histopathologic examination for the diagnosis.

References

1Rosenberg AE, Nielsen GP, Fletcher JA. Aneurysmal bone cyst. In: Fletcher CD, Unni KK, Mertens F, editors. WHO Classification of Tumors: Pathology and Genetics of Tumors of Soft Tissue and Bone. 3 rd ed. Lyon: IARC Press; 2005. p. 338-9.
2Jundt G. Aneurysmal bone cyst. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. WHO Classification of Tumors: Pathology and Genetics of Head and Neck Tumors. 3 rd ed. Lyon: IARC Press; 2005. p. 326.
3Bataineh AB. Aneurysmal bone cysts of the maxilla: A clinicopathologic review. J Oral Maxillofac Surg 1997;55:1212-6.
4Martins WD, Fávaro DM. Aneurysmal bone cyst of the coronoid process of the mandible: A case report. J Contemp Dent Pract 2005;6:130-8.
5Svensson B, Isacsson G. Benign osteoblastoma associated with an aneurysmal bone cyst of the mandibular ramus and condyle. Oral Surg Oral Med Oral Pathol 1993;76:433-6.
6Gadre KS, Zubairy RA. Aneurysmal bone cyst of the mandibular condyle: Report of a case. J Oral Maxillofac Surg 2000;58:439-43.
7López-Arcas JM, Cebrián L, González J, Burgueño M. Aneurysmal bone cyst of the mandible: Case presentation and review of the literatura. Med Oral Patol Oral Cir Bucal 2007;12:E401-3.
8Pelo S, Gasparini G, Boniello R, Moro A, Amoroso PF. Aneurysmal bone cyst located in the mandibular condyle. Head Face Med 2009;5:8.
9Matsuura S, Tahara T, Ro T, Masumi T, Kasuya H, Yokota T. Aneurysmal bone cyst of the coronoid process of the mandible. Dentomaxillofac Radiol 1999;28:324-6.
10Jaffe HL, Lichtenstein L. Solitary unicameral bone cyst with emphasis on the roentgen picture, the pathologic appearance and the pathogenesis. Arch Surg 1942;44:1004-25.
11James Ewing. Bone cystic lesions. In: En Ewing J de Saunders, editors. Neoplastic Diseases: A Treatise on Tumours. Philadelphia: Saunders Editores; 1940. p. 323-4.
12Panoutsakopoulos G, Pandis N, Kyriazoglou I, Gustafson P, Mertens F, Mandahl N. Recurrent t (16;17) (q22;p13) in aneurismal bone cysts. Genes Chromosomes Cancer 1999;26:265-6.
13Vicenzi G. Familial incidence in two cases of aneurysmal bone cyst. Ital J Orthop Traumatol 1981;7:251-3.
14Leithner A, Windhager R, Kainberger F, Lang S. A case of aneurysmal bone cyst in father and son. Eur J Radiol 1998;29:28-30.
15Kalantar Motamedi MH. Aneurysmal bone cysts of the jaws: Clinicopathological features, radiographic evaluation and treatment analysis of 17 cases. J Craniomaxillofac Surg 1998;26:56-62.
16Kiattavorncharoen S, Joos U, Brinkschmidt C, Werkmeister R. Aneurysmal bone cyst of the mandible: A case report. Int J Oral Maxillofac Surg 2003;32:419-22.
17Karabouta I, Tsodoulos S, Trigonidis G. Extensive aneurysmal bone cyst of the mandible: Surgical resection and immediate reconstruction. A case report. Oral Surg Oral Med Oral Pathol 1991;71:148-50.
18Capote-Moreno A, Acero J, García-Recuero I, Ruiz J, Serrano R, de Paz V. Giant aneurysmal bone cyst of the mandible with unusual presentation. Med Oral Patol Oral Cir Bucal 2009;14:E137-40.
19Peterson LJ, Indresano T, Marciani RD. Surgical management of benign nonodontogenic lesions of the jaws. Principles of Oral and Maxillofacial Surgery. Vol. 3. Philadelphia, Pennsylvania: Lippincott Philadelphia Editores; 1992. p. 733-40.
20Hernandez GA, Castro A, Castro G, Amador E. Aneurysmal bone cyst versus hemangioma of the mandible: Report of long-term follow-up of a self-limiting case. Oral Surg Oral Med Oral Pathol 1993;76:790-6.