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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 27  |  Issue : 3  |  Page : 479-483

Bilateral aneurysmal bone cyst of the mandible: A case report


1 Department of Oral Medicine and Radiology, YMT Dental College and Hospital, Navi Mumbai, Maharashtra, India
2 Department of Oral and Maxillofacial Surgery, Breach Candy Hospital, Mumbai, Maharashtra, India

Date of Submission08-Apr-2015
Date of Acceptance08-Apr-2015
Date of Web Publication25-Nov-2015

Correspondence Address:
Gopal HK Sharma
Department of Oral Medicine and Radiology, YMT Dental College and Hospital, Plot No. 18, Sector 4, Kharghar, Navi Mumbai - 410 210, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.170471

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   Abstract 

An aneurysmal bone cyst (ABC), usually considered a reactive lesion of bone rather than a cyst or true neoplasm, is believed to represent an exaggerated, localized, proliferative response of vascular tissue in bone. The case described here is of a 40-year-old female patient presenting with gradually increasing bilateral mandibular swelling of more than 1 year duration. The radiographic and cone beam computed tomography (CBCT) images showed bilateral, multilocular expansile radiolucent lesions in the mandible in the premolar-molar region. On considering the blood aspirate obtained and the histopathologic findings, the patient was diagnosed with bilateral aneurysmal bone cysts in the mandible, with probable pre-existing bone lesion.

Keywords: Aneurysmal bone cyst, bilateral aneurysmal bone cyst, cysts of jaws, jaws


How to cite this article:
Sharma GH, Dabir AV, Das DA, Talreja-Kanchan PP. Bilateral aneurysmal bone cyst of the mandible: A case report. J Indian Acad Oral Med Radiol 2015;27:479-83

How to cite this URL:
Sharma GH, Dabir AV, Das DA, Talreja-Kanchan PP. Bilateral aneurysmal bone cyst of the mandible: A case report. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2018 Dec 12];27:479-83. Available from: http://www.jiaomr.in/text.asp?2015/27/3/479/170471


   Introduction Top


An aneurysmal bone cyst (ABC), usually considered as a reactive lesion of bone rather than a cyst or true neoplasm, is believed to represent an exaggerated, localized, proliferative response of vascular tissue in bone. The etiology of ABCs remains unclear, and they are occasionally seen to develop in association with other primary lesions such as fibrous dysplasia, central hemangioma, giant cell granuloma, and osteosarcoma. [1] An ABC in the jaw usually manifests as an occasionally painful and a fairly rapidly enlarging bony swelling (usually buccal or labial). The involved area may be tender on palpation. Mandible is affected more often than maxilla (3:1) [2] with slight female predilection. ABCs rarely occur in jaws, with an incidence rate of 2% reported so far. Most of the cases have had a unilocular occurrence. But the rarity of the present case reported is its bilateral occurrence in the mandible.


   Case Report Top


A 40-year-old female presented with facial asymmetry due to bilateral swelling in the lower jaws [Figure 1]. She reported a gradually increasing mandibular swelling bilaterally which was slightly painful since about 1 year. Buccal cortical plate expansion was noted bilaterally in the premolar-molar region, obliterating the buccal vestibule [Figure 2]. Patient did not complain of any paresthesia or numbness. On electric pulp testing, all her teeth were vital, with no mobility present. No other significant intraoral findings were noticed. Her previous 1 year panoramic radiograph (OPG) [Figure 3] and cone beam computed tomography (CBCT) reports showed multilocular expansile lesions bilaterally in the body of the mandible [Figure 4], suggestive of a benign neoplastic process involving the mandible.
Figure 1: Facial asymmetry due to bilateral swelling of lower jaw, more prominent on the left side

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Figure 2: Intraoral image of obliteration of lower buccal vestibule bilaterally

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Figure 3: OPG (1 year before) showing large, bilateral, well-defi ned, multilocular radiolucency in the mandibular body

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Figure 4: A reconstructed panoramic CBCT image (1 year before) showing multilocular expansile lesion bilaterally in mandibular body region, of approximately 4.7 × 1.4 × 1.8 cm on the right side and 4.5 × 1.6 × 1.6 cm on the left side, in maximum anteroposterior, buccolingual, and superoinferior dimensions, respectively

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CBCT was again advised and large, destructive, predominantly radiolucent lesions were seen in the right and left body of the mandible, extending from the respective canine region to one cm distal to 47 anteroposteriorly. Superoinferiorly, the lesions extended from the mid-root levels of the teeth up to the inferior border of the mandible with well-defined corticated margins exhibiting a scalloped pattern, especially between the roots of the teeth [Figure 5]. The size was found to be 4.5 × 1.3 × 1.7 cm on the right side [Figure 6] and 4.6 × 1.4 × 1.6 cm on the left side [Figure 7], suggesting very slight changes in the 1 year duration. The interior of the lesions was radiolucent with few bony septae seen originating from the buccal and lingual cortices, giving it a multilocular appearance. Both the buccal and lingual cortices showed evidence of expansion and possible perforation by the lesion bilaterally [Figure 8]. The lesion appeared to have displaced both the left and right inferior alveolar canals inferiorly, near the inferior border of the mandible. The inferior border of the mandible appeared intact with no sign of any breach in its contour. Few trabeculae were noted in the mandibular anterior region, which could represent normal trabecular pattern or early involvement in the anterior region [Figure 9]. On the basis of these imaging findings, bilateral keratocystic odontogenic tumor (KCOT), bilateral ABCs, central giant cell granulomas, cystic changes in a fibro-osseous lesion, and ameloblastoma were considered as differential diagnoses.
Figure 5: A reconstructed panoramic CBCT image (current) showing large, destructive, predominantly hypodense lesions, with corticated scalloped margins, displacing the inferior alveolar canal inferiorly. Lower border of mandible is intact

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Figure 6: CBCT images — multiplanar and 3D reconstructed — depicting widest dimensions of lesion on the right side

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Figure 7: CBCT images — multiplanar and 3D reconstructed — depicting widest dimensions of lesion on the left side

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Figure 8: CBCT images of sections along the arch, from the right posterior to the left posterior region

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Figure 9: A reconstructed panoramic CBCT image showing probable early involvement in the anterior region

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On performing fine needle aspiration at the molar region, with blood aspirate obtained from left (1 cc) and right (3 cc) sides, a provisional/working diagnosis of bilateral ABC of the mandible was considered. No atypical or malignant cells were noted in the aspirate. Under local anesthesia, the lesion was accessed and curetted completely [Figure 10]. Histopathologic examination of the postoperative specimen showed slight fibro-osseous stroma with ossicles and ABC like changes were seen with the cavity filled with hemorrhagic material [Figure 11]. No surgical complications were experienced and on follow-up, healing was uneventful. The patient is currently on follow-up [Figure 12].
Figure 10: Surgical curettage using intraoral buccal approach, under local anesthesia

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Figure 11: Photomicrograph of postoperative specimen showing slight fibro-osseous stroma with ossicles (arrows in left image). ABC like changes are seen with cavity fi lled with hemorrhagic material (arrows in right image)

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Figure 12: Postoperative OPG taken 5 months later

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   Discussion Top


Aneurysmal bone cyst, first described by Jaffe and Lichtenstein in 1942, is a non-neoplastic bone lesion which has been reported to affect mainly the long bones of the skeleton. Only 2% of the lesions appear in the jaws. The World Health Organization (WHO) defines ABC as a benign intra-osseous lesion, characterized by blood-filled spaces of varying size associated with a fibroblastic stroma containing multinucleated giant cells, osteoid, and woven bone. The characteristic radiological features of ABC in the long bones are well documented as a well-defined expansile radiolucent lesion surrounded by a thin overlying cortex. In contrast, the descriptions of ABC in the jaws are conflicting and vary from mainly a unilocular radiolucency to a "ballooned out" multilocular radiolucency with a honeycomb or soap bubble appearance. [3]

The etiology of ABC remains unknown, and it is not known whether the lesion arises de novo or represents the result of some form of vascular accident in a pre-existing lesion. Most believe that ABCs are the result of a vascular malformation within the bone. Three theories concerning the cause and pathogenesis of ABC have been proposed: Possible traumatic origin resulting in subperiosteal or intramedullary hemorrhage with misguided reparative processes; altered hemodynamic state leading to a dilated congested vascular bed, causing resorption and erosion of bone with expansion of the lesion; and a secondary phenomenon that occurs in a primary osseous lesion. Panoutsakopoulos et al. described three cases of ABCs with chromosomal anomalies with band 16q22 being involved in all three patients. [4] Similar findings were reported by Herens et al. [5] A familial incidence of ABC has also been reported in the literature. [6]

Maxillomandibular ABCs are more common in the second to third decades of life. Presentation and clinical course may be extremely variable, characterized by either a slow-growing, inconspicuous lesion found on radiographic examination or a sudden, rapidly expanding lesion with resultant bony destruction and facial asymmetry mimicking malignancy or a false aneurysm. [6] Although this patient presented with the symptoms in her fifth decade of life, the onset of the lesion is not known, and perhaps the clinical course could be slow growing.

The radiographic features are not pathognomonic and are sometimes confusing. The lesion may appear as unilocular, multilocular, soap bubble, honeycomb, or moth-eaten radiolucency causing expansion, destruction of bone, perforation of the cortices, and herniation into the soft tissues, or an associated periosteal reaction with reactive new bone forming a peripheral sclerotic border, which in some cases is difficult to differentiate from a subperiosteal hematoma. The course of the ABC is often confusing, for it may range from a self-limited lesion to an aggressive, rapidly destructive lesion mimicking a malignancy. Pathologic fracture of the jaw has also been reported. [6] According to Kaffe et al., [3] ABC has a variable radiological appearance and should be considered in the differential diagnosis of any unilocular or multilocular radiolucent lesion of the jaws as well as any mixed radiolucent-radiopaque lesion.

In this case, the expansile multilocular lesion thinned the inferior border of mandible with expansion of the buccolingual cortices, but with no pathological fracture. However, bilateral occurrence of ABC appears to be extremely rare. ABC can also arise from a pre-existing bone lesion. Bieseker et al. suggested the development of ABC in a pre-existing lesion, [7] while El Deeb et al. reviewed 53 cases of which 21 were associated with a pre-existing lesion of bone. [8] The characterization of the cyst by replacement of bone with spongy fibro-osseous tissues and a locally destructive multicystic lesion filled with blood is attributed to circulatory disturbances that lead to locally increased venous pressure. The formation of ABC de novo is not in doubt, but the frequency with which it is associated with giant cell granuloma, fibrous dysplasia, and cementifying fibroma of the jaws tends to suggest an exaggerated vascular malformation in these lesions. [9]

ABC is a giant cell lesion within a fibroconnective tissue stroma with blood caverns or sinusoids and no epithelial lining. Thus, it is considered to be a non-neoplastic, and presumably reactive, bone lesion. [6] The majority of the cases respond well to surgical curettage, and the lesions subsequently fill with bone. Recurrence has been attributed to inadequate access and, thus, incomplete removal of the lesion. [2],[6]


   Conclusion Top


The ABC of the jaws represents an enigmatic pseudocyst with variable clinicopathological, radiographic, and histological presentations. The bilateral expansile occurrence with the epicenter in the molar region, observed in this case, could be a slow-growing lesion with a long clinical course. Since the histological appearance was predominantly of ABC, we consider this case to be a primary case of ABC. This case was unique because of its bilateral occurrence.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
White SC, Pharoah MJ. Diseases of bone manifested in jaws. In: White SC, Pharoah MJ, editors. Oral Radiology: Principles and Interpretation. 6 th ed. Saint Louis, MO: Mosby; 2009. p. 445-6.  Back to cited text no. 1
    
2.
Wood NK, Goaz PW. Differential Diagnosis of Oral and Maxillofacial Lesions. 5 th ed. St. Louis: Mosby; 1997. p. 343-54.  Back to cited text no. 2
    
3.
Kaffe I, Naor H, Calderon S, Buchner A. Radiological and clinical features of aneurysmal bone cyst of the jaws. Dentomaxillofac Radiol 1999;28:167-72.  Back to cited text no. 3
    
4.
Panoutsakopoulos G, Pandis N, Kyriazoglou I, Gustafson P, Mertens F, Mandahl N. Recurrent t(16;17)(q22;p13) in aneurysmal bone cysts. Genes Chromosomes Cancer 1999;26:265-6.  Back to cited text no. 4
    
5.
Herens C, Thiry A, Dresse MF, Born J, Flagothier C, Vanstraelen G, Allington N, Bex V. Translocation (16;17)(q22;p13) is a recurrent anomaly of aneurysmal bone cysts. Cancer Genet Cytogenet 2001;127:83-4.  Back to cited text no. 5
    
6.
Motamedi MH, Navi F, Eshkevari PS, Jafari SM, Shams MG, Taheri M, et al. Variable presentations of aneurysmal bone cysts of the jaws: 51 cases treated during a 30-year period. J Oral Maxillofac Surg 2008;66:2098-103.  Back to cited text no. 6
    
7.
Biesecker JL, Marcove RC, Huvos AG, Miké V. Aneurysmal bone cysts. A clinicopathologic study of 66 cases. Cancer 1970;26:615-25.  Back to cited text no. 7
    
8.
El Deeb M, Sedano HO, Waite DE. Aneurysmal bone cyst of the jaws. Report of a case associated with fibrous dysplasia and review of the literature. Int J Oral Surg 1980;9:301-11.  Back to cited text no. 8
[PUBMED]    
9.
Saheeb BD, Ojo M, Obuekwe ON. Aneurysmal bone cyst: A primary or secondary lesion? Niger J Clin Pract 2007;10:243-6.  Back to cited text no. 9
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12]



 

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