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CASE REPORT
Year : 2010  |  Volume : 28  |  Issue : 4  |  Page : 307-310
 

Maxillary aneurysmal bone cyst: Report of a rare case


Department of Pedodontics and Preventive Dentistry, KM Shah Dental College and Hospital, Piparia, Waghodia, Vadodara, Gujarat, India

Date of Web Publication25-Jan-2011

Correspondence Address:
M B Sheth
26, Gajanand Soc., Manjalupur Naka, Vadodara - 390 011, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.76164

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   Abstract 

Everyone likes to see, hear, and know about rare things. An aneurysmal bone cyst (ABC) is an uncommon non-neoplastic lesion of the bone, mostly affecting long bones and spine. This rare jaw lesion usually affects the mandible but origin from the maxillary anterior region is even rarer. This paper describes a case of an ABC in an 8-year-old female child with a progressively increasing swelling in the right nasomaxillary region developed over a period of 1.5 months. Treatment consisted of the excision and enucleation of the lesion through an intraoral approach under general anesthesia. The higher prevalence in young adulthood has made the role of pedodontists vital in diagnosing ABCs, at the earliest.


Keywords: Aneurysmal bone cyst, benign tumor, enucleation


How to cite this article:
Sheth M B, Sujan S G, Poonacha K S. Maxillary aneurysmal bone cyst: Report of a rare case. J Indian Soc Pedod Prev Dent 2010;28:307-10

How to cite this URL:
Sheth M B, Sujan S G, Poonacha K S. Maxillary aneurysmal bone cyst: Report of a rare case. J Indian Soc Pedod Prev Dent [serial online] 2010 [cited 2019 Oct 23];28:307-10. Available from: http://www.jisppd.com/text.asp?2010/28/4/307/76164



   Introduction Top


The World Health Organization defines an aneurysmal bone cyst (ABC) as "a benign tumor like lesion with an expanding osteolytic nature, consisting of blood filled spaces of variable sizes separated by connective tissue septae containing trabeculae or osteoid tissue and osteoclastic giant cells." [1],[2] Jaffe chose the name "aneurysmal" to emphasize the "blown out," distended contour of the affected bone, and the words "bone cyst" to underscore that when the lesion is entered through a thin shell of bone, it appears largely as a blood-filled cavity. [3] An ABC usually affects long bones of the body and its involvement of facial bones is relatively rare.

This article adds one more aspect in the diversity of clinical and biological behavior of ABCs in the maxillofacial region as here the lesion occurred in maxilla and in the first decade of life which makes it a rare case.


   Case Report Top


An 8-year-old female patient reported with the chief complaint of facial asymmetry due to progressive swelling in the right side of the face since 1.5 months. The patient gave a history of impact with a rubber ball on the right side of her face while playing. There was a history of severe post-traumatic pain and swelling but not of unconsciousness or bleeding from the ear, nose, and oral cavity.

On extraoral examination, there was a smooth diffuse hard swelling of about 4 Χ 5 cm in the right nasomaxillary region [Figure 1]. The swelling extended superiorly from the medial one-third of the infraorbital margin to the upper vermilion border inferiorly and medially up to the nasal septum. The nasal septum was deviated to the left side, the nasolabial sulcus obliterated, and the right angle of mouth lowered. The swelling was firm and tender with eggshell crackling on palpation. There was no murmur or bruit on auscultation. Intraorally, vestibular obliteration with respect to maxillary right incisors was observed [Figure 2]. The maxillary right central incisor was partially extruded and the right lateral incisor was displaced labially. Palatal swelling was seen in the anterior region. There was no pus discharge or sinus present and the mucoperiosteal tissue was intact.
Figure 1 :Preoperative extraoral view showing the swelling and facial asymmetry on the right side of the face

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Figure 2 :Preoperative intraoral view showing maxillary right vestibular obliteration

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The electrical and thermal tests revealed both maxillary right incisors vital. The maxillary occlusal radiograph exhibited a definite radiolucent lesion extending from the maxillary right central incisor to the right deciduous first molar [Figure 3]. An orthopantomograph showed a unilocular radiolucency without scalloping that resembled blown out distension [Figure 4]. Aspiration with an 18 G needle revealed dark-colored blood.
Figure 3 :Maxillary occlusal radiograph showing the radiolucent lesion

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Figure 4 :Orthopantamograph showing blown out unilocular radiolucency

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Considering the recent history of trauma, clinical features, radiographic investigations and absence of bruits, thrills, and pulse pressure, the lesion was provisionally diagnosed as ABC. Considering the size and extent of the lesion, it was decided to surgically excise and enucleate the cyst under general anesthesia.

An intraoral crevicular incision was placed from the mesial surface of the right maxillary central incisor to the mesial surface of the right deciduous first molar. The mucoperiosteal flap was raised. A bluish red sponge-like lesion was explored on removing the outer cortical plate. The lesion was enucleated [Figure 5]. All the teeth and tooth buds involved in the lesion were removed (maxillary right lateral incisor, deciduous canine and first molar, and tooth buds of permanent canine and first premolar) [Figure 6]. The margins of the bone were trimmed and the incision was closed with the 3-0 catgut suture material [Figure 7].
Figure 5 :Intraoperative view of the enucleation of the cyst

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Figure 6 :Teeth removed intraoperatively

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Figure 7 :Sutures placed after enucleation

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The lesion resembled blood-soaked sponge [Figure 8] and was sent for histopathological examination which showed numerous variably sized blood-filled spaces separated by fibrous septae containing spindle-shaped cells and scattered multinucleated giant cells suggestive of an ABC [Figure 9].
Figure 8 :Section of the excised lesion resembling blood-soaked sponge

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Figure 9 :Microscopic image of the lesion

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The patient's postoperative course was uneventful and the wound healed normally. The facial asymmetry was corrected [Figure 10] and the patient was rehabilitated with a removable partial prosthesis for functional and esthetic needs.
Figure 10 :Postoperative image showing the facial asymmetry corrected

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   Discussion Top


The ABCs are benign neoformations which can affect all the skeletal bones. ABCs occur very rarely in the jaws. Considering all types of jaw cysts, the ABC is extremely rare with 0.5% predilection. About two-thirds of cases have been reported in the mandible and only one-third in the maxilla. [4],[5],[6] They represent about 1.5% of all nonodontogenic and nonepithelial cysts of the jaws. The normal age of occurrence is 13-15 years and 80% of patients are less than 20 years old with no gender predilection. [4] The ABCs can occur in solid, vascular, or mixed form. [7]

The etiology of ABCs is unclear and controversial. The ABC is most common in those regions of the skeleton where there is both a relatively high venous pressure and high marrow content. This explains the rarity of ABCs in the skull bones where there is low venous pressure. Etiopathogenetically, it is thought to be either a neoplasm, a developmental anomaly, response to trauma, chronic infection, arteriovenous anomaly, degenerative lesion, or subperiosteal intraosseous hematoma. [2],[6]

Preoperative diagnosis of ABCs can be difficult because of the similarity in clinical presentation to other lesions like traumatic bone cyst, giant cell tumor, hyperparathyroidism, fibrous dysplasia, desmoplastic fibroma, fibrous histiocytoma, hemangioma, osteogenic sarcoma, and adenomatoid odontogenic tumor. [8] The aspiration of blood from the lesion should give suspicion of a vascular lesion or an ABC. The absence of bruits, thrills, and pulse pressure helps to clinically differentiate an ABC from a vascular lesion. Definitive diagnosis can be made only after excisional biopsy.

Depending on the size, site, and extent of the lesion, the treatment options range from curettage, enucleation, percutaneous sclerotherapy, diagnostic and therapeutic embolization, block resection and reconstruction, and systemic calcitonin therapy. Self-healing cases have also been reported on long-term follow-up. [9] Conventional treatment has been directed at the surgical removal of the entire lesion, or of the affected area as much as possible. Recurrence is seen in 10-44% of cases, with 90% recurring within 2 years. The use of radiation has not been advocated as it may fail to arrest the lesion and can result in sarcomatous changes. [10]

In the present case, enucleation was the surgical choice as the tumor was well encapsulated and the careful inspection of the surgical bed showed no signs of tumor fragments or adherences to neighboring structures.


   Acknowledgment Top


We are grateful to Dr. Kiran Desai, Head of the Department of Oral and Maxillofacial Surgery, KM Shah Dental College and Hospital, Piparia.

 
   References Top

1.Motamedi MH, Yazdi E. Aneurysmal bone cyst of the jaws: Analysis of 11 cases. J Oral Maxillofac Surg 1994;52:471-5.   Back to cited text no. 1
[PUBMED]  [FULLTEXT]  
2.Struthers P, Shear M. Aneurysmal bone cyst of the jaws: Part II: Pathogenesis. Int J Oral Surg 1984;13:92-100.  Back to cited text no. 2
    
3.Jaffe HL. Aneurysmal bone cyst. Bull Hosp Joint Dis 1950;11:3-13.  Back to cited text no. 3
[PUBMED]    
4.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. 4
[PUBMED]  [FULLTEXT]  
5.Segall L, Cohen-Kerem R, Ngan BY, Forte V. Aneurysmal bone cysts of the head and neck in pediatric patients: A case series. Int J Pediatr Otorhinolaryngol 2008;72:977-83.   Back to cited text no. 5
[PUBMED]  [FULLTEXT]  
6.Tillman BP, Dahlin DC, Lipscomb PR, Stewart JR. Aneurismal bone cyst: An analysis of 95 cases. Mayo Clin Proc 1968;43:478-95.  Back to cited text no. 6
[PUBMED]    
7.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. 7
[PUBMED]  [FULLTEXT]  
8.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. 8
[PUBMED]  [FULLTEXT]  
9.Malghem J, Maldague B, Esselinck XW, Noel H, De Nayer P, Vincent A. Spontaneous healing of aneurysmal bone cysts: A report of three cases. J Bone Joint Surg Br 1989;71:645-50.  Back to cited text no. 9
    
10.Cohan W. Sacrcoma arising in irradiated bone. Cancer 1984;1:3.  Back to cited text no. 10
    


    Figures

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


This article has been cited by
1 Aneurysmal bone cyst of maxilla- A rare case report and review
Mohammed, R.B. and Shirisha Rani, G. and Chowdary, M.S. and Shereen, A. and Yellarti, P.K.
Journal of International Dental and Medical Research. 2013; 6(1): 49-53
[Pubmed]



 

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