Home | About us | Editorial board | Ahead of print | Current issue | Archives | Search | Submit article | Instructions | Subscribe | Advertise | Contact us |  Login 
National Journal of Maxillofacial Surgery
 
Print this page Email this page Small font sizeDefault font sizeIncrease font size
Users Online: 458
 


 
Table of Contents
CASE REPORT
Year : 2013  |  Volume : 4  |  Issue : 1  |  Page : 107-110  

Giant aneurysmal bone cyst of the mandible: A case report and review of literature


Department of Oral and Maxillofacial Surgery, NIMS Dental College, Shobha Nagar, Jaipur, Rajasthan, India

Date of Web Publication7-Sep-2013

Correspondence Address:
Gaurav Bharadwaj
NIMS Dental College, Shobha Nagar, Jaipur-Delhi National Highway, Jaipur, Rajasthan
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-5950.117819

Rights and Permissions
   Abstract 

Aneurysmal bone cysts are rare benign lesions of bone tissue. They are composed of vascular spaces blood-filled and surrounded by fibrous tissue septa. They are considered as pseudo cysts because of lack of epithelial lining. Here, we describe a giant case of ABC in 12-year-old female child having a massive swelling over the right side of the mandible treated with segmental resection and reconstruction with a reconstruction plate. Case is also discussed with the review of literature.

Keywords: Aneurysmal bone cysts, benign bone lesions, mandible, pseudocysts


How to cite this article:
Bharadwaj G, Singh N, Gupta A, Sajjan AK. Giant aneurysmal bone cyst of the mandible: A case report and review of literature. Natl J Maxillofac Surg 2013;4:107-10

How to cite this URL:
Bharadwaj G, Singh N, Gupta A, Sajjan AK. Giant aneurysmal bone cyst of the mandible: A case report and review of literature. Natl J Maxillofac Surg [serial online] 2013 [cited 2019 Nov 22];4:107-10. Available from: http://www.njms.in/text.asp?2013/4/1/107/117819


   Introduction Top


Jaffé and Lichtenstein were first to recognize ABC affecting the metaphyseal region of long bones. Berneir and Bhaskar described the first case of ABC in 1958. [1],[2] Fifty percent of ABC arise in long bones 20% in vertebral column and 1.5% in mandible. [3],[4] It affects young person under 20 year of age with no gender predilection. [4],[5]

It can be classified into three types. Conventional or vascular types manifests as a rapidly growing expansive, destructive lesion causing cortical perforation and soft tissue invasion. The solid type may present as a small asymptomatic lesion first noticed as a small asymptomatic lesion on a routine radiograph. [6],[7] A third form or mixed variant demonstrates 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. [7]

These are nonneoplastic but locally aggressive tumor with occasional rapid growth, which should be differentiated from other multilocular processes like ameloblastoma, ossifying fibroma, epithelial cyst, giant cell granuloma. [1]


   Case Report Top


A huge mandibular swelling six months before started as a small swelling and gradually progressed to present size, her medical and family history was remarkable. On extraoral examination there was facial asymmetry with apparent massive swelling involving the right side of the lower jaw measuring approx. 6.8 * 7.2 cm. The swelling was firm and non tender [Figure 1].
Figure 1: Pre operative extraoral photograph

Click here to view


Intra oral examination reveals a diffuse swelling in relation to the right lower first premolar, second premolar and first and second molar with displacement of first and second molar, there was cortical plate expansion and erosion on the both the sides with huge soft tissue mass obliterating the buccal, as well as the lingual sulcus pushing the tongue to the contra lateral side. The superior surface of the mass was in the contact with maxillary posterior teeth, which were causing ulceration on the superior surface of the mass [Figure 2].
Figure 2: Pre operative intraoral photograph

Click here to view


The child had a very poor oral hygiene with drooling of the saliva and difficulty in mastication, on aspiration rapid filling of the syringe with frank blood took place there was no bruit on auscultation.

A panoramic radiograph [Figure 3] revealed a large multilocular radiolucency involving the right body of mandible extending from distal of first molar and extending on to the ramus involving the inferior border of the mandible completely, and going up to the coronoid process and subcondylar region.
Figure 3: Pre operative OPG

Click here to view


MRI of the lesion revealed a large lobulated expansile mass with central necrotic hemorrhagic areas measuring 6.8 * 7.7 * 6.8 cm in anteroposterior, transverse and superoinferior dimension. Multiple vascular flow voids in the lesion arising from right ECA, lingual and facial artery [Figure 4].
Figure 4: Pre operative MRI

Click here to view


In view of the large size of the lesion, treatment plan of segmental resection was made, after ensuring patient fitness for GA, the patient was taken for segmental resection under fiber optic NET intubation, an extended neck crease incision was given and the lesion was exposed in its entirety ECA was dissected out and a loop was kept ready to be used in case of extensive hemorrhage.

The mass was excised with safe margin and was reconstructed with Titanium Angled Reconstruction plate.

Gross examination of the mass revealed the cortical expansion with erosion and replacement of endosteal tissue with large dilated blood filled spaces with tissue necrosis and evidence of incompletely osteoid tissue. There was no significant blood loss after surgery.

The tissue was send for histopathological examination, microscopic examination reveals large lined by endothelial cells and there was no presence of giant cells [Figure 5].
Figure 5: Histopathological photo

Click here to view


Patient made an uneventful recovery [Figure 6].
Figure 6: Post operative extraoral photograph

Click here to view


Reconstruction plate was well contoured with the mandible [Figure 7].
Figure 7: Post operative OPG

Click here to view



   Discussion Top


The term aneurysmal is a misnomer and refers to the blow out effect or expansion of the affected bone that appears in this type of lesions. [8]

It is a pseudo cyst lacking epithelial lining, [1],[5],[8] it comprises the 5% of all the lesion of craniofacial and maxillofacial bones. [2]

The etiopathogenesis of ABC is highly debatable and controversial and many theories have explained their origin, Jaffé and Lichtenstein have suggested a vascular origin in which there is alteration in local hemodynamics causing increased venous pressure and engorgement of the vascular bed leading to the resorption. [1],[9]

Steiner and Kantor have suggested that ABC can develop as primary or secondary lesion associated with other bone diseases, Struthers and Shear have also concluded that ABC can occur as a secondary phenomenon in a preexisting lesion and giant cell granuloma. [3]

Levy et al., has proposed a history of trauma and subperiosteal hematoma formation is a essential factor in the development of ABC, however, Tillman et al., have reported 95 cases with no history of trauma. [7]

Hernandez et al., classified ABC as primary and secondary, primary could be congenital or acquired. The congenital is seen in children and young adults with no history of trauma whereas the acquired is found in adults with history of trauma. The secondary is postulated to be associated with degeneration of preexisting lesion such as cyst, tumor or fibro osseous lesions. [3]

The two lesion can exist independently, hence, ABC is considered as non neoplastic fibro-dysplastic, non cystic bone entity. [3],[10]

In our case also there was no history of trauma so etiology could be either due to alteration in local hemodynamics or degeneration of any preexisting lesion at the involved site.

Panoutsakopoulos et al., had described three cases with chromosomal anomalies involving band 16q22.

Familial incidence has been reported in the literature. [11],[12],[13]

ABC are most commonly found in long bones and in vertebral column 1.9% reported in jaws and unusual location like condyle and coronoid process been reported. [3],[7],[8],[14]

It is extremely variable in clinical presentation ranging from small lesion to rapidly growing expansible destructive lesion causing deformity, pain and cortical perforation.

The latter presentation was found in our case also where there was rapidly expansile growth with the short history.

The radiological features are quite conflicting, it may appear radiolucent, radio-opaque or mixed. The bone can appear expanded, cystic resembling a honey comb or soap bubble in appearance similar to what is seen in benign odontogenic tumors and diagnosis. Based on radiographic appearance impossible because there are multiple lesions having similar radiographic appearance such as Ameloblastoma, Myxoma, Central giant cell tumor and central hemangiomas. [15]

Our lesion also had a multilocular appearance resembling other odontogenic lesions.

MRI features

MRI is mandatory in complex cases such as the reported case in order to improve the plain radiographic examination and CT scans as it is more accurate in soft tissue contrast.

MRI finding of fluid filled levels inside the lesion are highly specific of ABC. [16]

In our case also MRI clearly depicted the flow voids arising from right ECA, lingual and facial arteries.

Angiography is sometimes used when MRI shows hypervascularization. [8]

Histologically, ABC consists of blood filled spaces separated by fibrous septa with multinucleated giant cells and osteoid. Hemosiderin is present in variable amount and there is evidence of osteoid and bone formation, this is characteristic of classic or vascular form.

The histopathological feature in our case was consistent with the above mentioned features, however, we did not find presence of any giant cells in our lesion.

Solid form is the other type which is a non-cystic variant with solid grey white tissue with osteoid and calcifying fibro-myxoid tissue. The mixed form demonstrates elements of both vascular and solid type. [8]

Treatment usually directs to complete removal of the lesion, the modalities are enucleation and curettage. Diagnostic and therapeutic embolization, segmental resection and reconstruction. Simple curettage is associated with high recurrence but few authors have reported no recurrence following simple curettage of mandibular lesion. [1],[17]

Segmental resection must be done in cases of multiple recurrence or extension to the overlying tissue. [1],[8],[14]

Recurrence rate ranges from 20-30% and seem to occur more frequently, first year after surgery. [18]

Self healing cases have also been reported on long term follow-up. [19]

Several author recommends immediate reconstruction of the defect with autografts in cases of aesthetic deformity high-risk of fracture and loss of mandibular continuity. [3],[5],[14]

The present case was treated by segmental resection and reconstruction by a reconstruction plate due to the size and the extension of the lesion reconstruction, with free tissue transfer was planned for a later date.


   Conclusion Top


A case of giant ABC of mandible in a 12-year-old female child treated segmental resection and reconstruction of the defect with a reconstruction plate, is presented along with review of literature.

 
   References Top

1.Motamedi MH. Destructive aneurysmal bone cyst of the mandibular condyle: Report of a case and review of the literature. J Oral Maxillofac Surg 2002;60:1357-61.  Back to cited text no. 1
[PUBMED]    
2.Kalantar Motamedi MH. Aneurysmal bone cyst of the jaws: Clinicopathological features, radiographic evaluation and treatment analysis of 17 cases. J Craniomaxillofac Surg 1998;26:56-62.  Back to cited text no. 2
[PUBMED]    
3.Gadre KS, Zubairy RA. Aneurysmal bone cyst of the mandibular condyle: Report of a case. J Oral Maxillofac Surg 2000;58:439-43.  Back to cited text no. 3
[PUBMED]    
4.Goyal A, Tyagi L, Syal R, Agarwal T, Jain M. Primary aneurismal bone cyst of coronoid process. BMC Ear Nose Throat Disord 2006;6:4.  Back to cited text no. 4
    
5.Kiattavorncharoen 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.  Back to cited text no. 5
[PUBMED]    
6.López-Arcas Calleja JM, Cebrián Carretero JL, González Martín J, Burgueño M. Aneurysmal bone cyst of the mandible: Case presentation and review of the literature. Med Oral Patol Oral Cir Bucal 2007;12:E401-3.  Back to cited text no. 6
    
7.Pelo S, Gasparini G, Boniello R, Moro A, Amoroso PF. Aneurysmal bone cyst located in the mandibular condyle. Head Face Med 2009;5:8.  Back to cited text no. 7
[PUBMED]    
8.Capote-Moreno A, Acero J, Garcia-Recuero I, Ruiz J, Serrano R, de Paz V. Giant aneurismal bone cyst of the mandible with unusual presentation. Med Oral Patol Oral Cir Bucal 2009;14:E137-40.  Back to cited text no. 8
    
9.Jaffé HL, Lichtenstein L. Solitary unicameral bone cyst. With emphasis on roentger picture, the pathological appearance and the pathogenesis. Arch Surg 1942;44:1004-25.  Back to cited text no. 9
    
10.Eisenbud L, Attie J, Garlick J, Platt N. Aneurysmal bone cyst of the mandible. Oral Surg Oral Med Oral Pathol 1987;64:202-6.  Back to cited text no. 10
[PUBMED]    
11.Vicenzi G. Familial incidence in two cases of aneurismal bone cysts. Ital J Orthop Traumatol 1981;7:251-3.  Back to cited text no. 11
[PUBMED]    
12.Power RA, Robbins PD, Wood DJ. Aneurysmal bone cyst in monozygotic twins: A case report. J Bone Joint Surg Br 1996;78:323-4.  Back to cited text no. 12
[PUBMED]    
13.Leithner A, Windhager R, Kainberger F, Lang S. A case of aneurysmal bone cyst in father and son. Eur J Radiol 1998;29:28-30.  Back to cited text no. 13
[PUBMED]    
14.Martins WD, Fávaro DM. Aneurysmal bone cyst of the coronoid process of the mandible: A case report. J Contemp Dent Pract 2005;6:130-8.  Back to cited text no. 14
    
15.Karabouta I, Tsodoulos S, Trigonidis G. Extensive aneuresymal bone cyst of the mandible: Surgical resection and immediate reconstruction. Oral Surg Oral Med Oral Pathol 1991;71:148-50.  Back to cited text no. 15
[PUBMED]    
16.Asaumi J, Konouchi H, Hisatomi M, Matsuzaki H, Shigehara H, Honda Y, et al. MR features of aneurysmal bone cyst of the mandible and characteristics distinguishing it from other lesions. Eur J Radiol 2003;45:108-12.  Back to cited text no. 16
    
17.Devi P, Thimmarasa VB, Mehrotra V, Agarwal M. Aneurysmal bone cyst of the mandible: A case report and review of literature. J Oral Maxillofac Pathol 2011;15:105-8.  Back to cited text no. 17
[PUBMED]  Medknow Journal  
18.Panoutsakopoulos 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.  Back to cited text no. 18
[PUBMED]    
19.Malghem J, Maldague B, Esselinckx W, Noel H, De Nayer P, Vincent A. Spontaneous healing of aneurismal bone cysts: A report of three cases. J Bone Joint Surg Br 1989;71:645-50.  Back to cited text no. 19
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]


This article has been cited by
1 Bimaxillary Aneurismal Bone Cyst in Patient with End Stage Renal Disease and Hyperparathyroidism: A Rare Case Report and Review of the Literature
Sedigheh Bakhtiari,Mahin Bakhshi,Fatemeh Mashhadiabbas,Hasan Mir Mohammad Sadeghi,Zahra Elmi Rankohi,Somayeh Rahmani
Case Reports in Dentistry. 2016; 2016: 1
[Pubmed] | [DOI]
2 Palatal Swelling: A Diagnostic Enigma
Ramalingam Suganya,Narasimhan Malathi,Harikrishnan Thamizhchelvan,Subramaniam Ramkumar,G. V. V. Giri
Case Reports in Dentistry. 2016; 2016: 1
[Pubmed] | [DOI]
3 Aneurysmal bone cyst of the mandible: A case report
Shina Mohd Ariffin,Noraini Nun Nahar Yunus
Pediatric Dental Journal. 2014;
[Pubmed] | [DOI]



 

Top
   
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed4416    
    Printed47    
    Emailed1    
    PDF Downloaded459    
    Comments [Add]    
    Cited by others 3    

Recommend this journal