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Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
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Year : 2012  |  Volume : 30  |  Issue : 4  |  Page : 340-342

Aneurysmal bone cyst of maxilla existing concurrently with a giant cell granuloma of mandible: A case report of an unusual presentation

1 Department of Oral and Maxillofacial Surgery, Swami Devi Dyal Hospital and Dental College, Barwala, India
2 Department of Dentistry Agroha Medical College, Agroha, India
3 Department of Prosthodontics, Swami Devi Dyal Hospital and Dental College, Barwala, India

Date of Web Publication19-Mar-2013

Correspondence Address:
S S Arora
44-B, Model Town, Ambala - 134 003, Haryana
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0970-4388.108938

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Aneurysmal bone cysts (ABCs) are relatively rare but well-described expansile, osteolytic psuedocysts are most commonly seen in the long bones, pelvis, and vertebra. Although a well-described lesion, much confusion exists in its pathogenesis especially of the associations of this lesion with other lesions. Approximately, one-third of patients with ABC present simultaneously with another bone pathology. The aim of this paper is to describe a rare case of simultaneously occurring ABC of the maxilla and mandible with the mandibular lesion occurring predominantly as a giant cell granuloma. The paper discusses the various histopathological variations of ABC as well as different lesions associated with ABC and tries to provide a review of different pathogenesis theories of this interesting lesion.

Keywords: Aneurysmal bone cysts, aneurysmal bone cysts, giant cell granuloma

How to cite this article:
Arora S S, Grover A, Kumar V V, Arora S. Aneurysmal bone cyst of maxilla existing concurrently with a giant cell granuloma of mandible: A case report of an unusual presentation. J Indian Soc Pedod Prev Dent 2012;30:340-2

How to cite this URL:
Arora S S, Grover A, Kumar V V, Arora S. Aneurysmal bone cyst of maxilla existing concurrently with a giant cell granuloma of mandible: A case report of an unusual presentation. J Indian Soc Pedod Prev Dent [serial online] 2012 [cited 2021 Jul 31];30:340-2. Available from: https://www.jisppd.com/text.asp?2012/30/4/340/108938

   Introduction Top

Aneurysmal bone cyst (ABC) was first described by Jaffe and Lichtenstein in 1942. [1] Bernier and Bhaskar are credited with the first case report of two patients having an ABC involving the mandible. [2] WHO classifies ABC as a tumor-like lesion. It defines it as "An expanding osteolytic lesion consisting of blood filled spaces of variable sizes separated by connective tissue septa containing trabeculae of osteoid tissue and osteoclast giant cells." [3] ABCs are more commonly found in the long bones (50%) and the vertebral column (20%). Only 1.9% of all ABCs occur in the jaws representing 1.5% of all non-odontogenic cysts. [4] ABCs exist in two clinicopathological forms-as a primary or as a secondary lesion arising from another osseous condition-it includes fibrous dysplasia, cementifying fibroma, giant cell granuloma, and certain unspecified lesions. [5],[6] Histologically, it can be of three variants: (1) vascular type, (2) solid type, and (3) mixed type. [7],[8]

This case report is of a 6-year-old boy presenting with an ABC of the maxilla occurring concurrently with a giant cell lesion of the mandible showing aneurysmal cyst changes. The aim of this paper is to highlight this unusual presentation and stresses on the need for close monitoring of these lesions to record the transition of one to the other.

   Case Report Top

A 6-year-old male patient presented with a complaint of an asymptomatic swelling in the left cheek and lower jaw region since 6 months which had increased to the present size. Medical records and family history were unremarkable and there was no history of trauma. On examination, there was a well-defined swelling in the left upper jaw and lower jaw. The upper lesion measured 3 cm × 2 cm and the lower one measured 2 cm × 2 cm. The swelling was non-tender and firm. Intraoral examination showed an obliterated vestibule in the region of left upper deciduous central incisor to the first permanent molar and the lower one from the distal of canine to the first permanent molar. On aspiration, blood was seen to fill the syringe from the maxillary lesion. However, the mandibular lesion was negative to aspiration.

Computed tomography scans of the patient demonstrated a ballooning destruction of the maxilla with a thin outline of sub-periosteal bone. There was an extension of the lesion medially causing narrowing of the nasal cavity. The mandibular lesion caused expansion of both the cortex.

Complete curettage of both the lesions was done under general anesthesia and the tissue was sent for histopathological examination. The maxillary lesion revealed numerous small vascular spaces lined by endothelial cells. Pools of red blood cells' hemosiderin pigment were seen along with giant cells suggestive of ABC. The mandibular lesion contained multinucleated giant cells in a vesiculated fibroblast connective tissue stroma. Presence of hemosiderin pigment was also seen. In certain sections, there were small vascular spaces along with giant cells suggestive of an ABC.

   Discussion Top

A case of ABC of maxilla and central giant cell granuloma of the mandible showing aneurysmal cyst-like changes in the mandible of a 6-year-old boy is described.

ABCs are more common in bones with high venous pressure and marrow content like the long bones. [9] ABCs when present in skull bones are more common in the mandible when compared to maxilla (3:1). [4],[10] The etiology and pathogenesis of ABC remain obscure. Jaffe and Lichtenstein [1] proposed that the ABC often arises from a pre-existing lesion of bone most of which has been destroyed by hemorrhage. Other authors [10] proposed the cause to be vascular disturbance, which maybe in the form of a venous thrombosis or arteriovenous aneurysm; the increased pressure of this disturbance may result in a dilated, engorged intra bony venous bed. The presence of giant cells in a fibrous matrix represents a physiological process of resorption and replacement rather than a primary lesion. In 1978, Hillerup and Hjorting-Hansen [11] proposed the theory that ABC, central giant cell granuloma, and simple bone cyst arise from some vascular defect and that trauma or some form of aneurysm leads to the formation of an intramedullary hematoma. When this hematoma is in contact with a large vessel (e.g., inferior alveolar bundle), ballooning via blood pressure occurs and an ABC results.

The reactive nature of ABCs has been challenged by the works of Pauontsakoponlos et al. [12] who demonstrated that primary ABCs exhibited chromosomal translocation t(16;17)(q22;p13) as a recurrent cytogenetic abnormality. Those findings were later confirmed by Dal Cin et al. [13] These findings highlight that the primary and secondary ABCs represent different concepts; gene translocation leading to primary ABCs and secondary ones arising from a pre-existing lesion.

In their review of 66 cases of ABCs, Biesecker et al. [14] found an association with ossifying fibroma, central giant cell granuloma, fibromyxoma, chondroblastoma, osteoblastoma, and unicameral bone cyst. They concluded that Central giant cell granuloma and ABC can coexist or ABC and CGCG can occur concurrently with transition of CGCG into ABC at some stage. According to a recent report by Krausdorf and Sweet, [15] an ABC should be considered only as a secondary lesion; the pre-existing pathology in the primary lesion may have been overlooked or the morphological changes into ABC might have erased any traces of the pre-existing lesion. In their analysis of 17 cases of ABCs of the Jaws, Zhao et al. [16] concluded that most of the ABCs were secondary in nature and confirmed that they were frequently combined with ossifying fibroma.

Although no gene studies were conducted on our patient, the mandibular lesion was a reactive one co-existing with a giant cell granuloma.

Histologically, primary ABCs contain fibrous connective tissue stroma or septae between numerous blood-filled caverns or sinusoids. This is the vascular form which presents as a spongy blood-filled lesion clinically. Young fibroblasts and giant cells are abundant which is similar to the central giant cell granuloma which is, however, devoid of vascular caverns. [10],[17] The solid variant is, however, similar to a central giant cell granuloma where the vascular spaces are small lined by a cellular fibrous tissue. In these, the prominent feature is a fibro-histiocytic proliferation with scattered osteoclast-like giant cells. Osteoblastic differentiation with osteoid and foci of a calcifying fibromyxoid tissue would complete the histological picture. [10],[15] The mixed variant of ABC has features of both vascular and solid types and this probably represents a transitional phase of the lesion.

In our case, the maxillary lesion represented a typical ballooning clinical appearance and histologically a vascular ABC. The mandibular lesion represented a central giant cell granuloma showing aneurismal cyst-like changes in one or two sections.

To our knowledge in the review of literature of ABC, this is the first report of a primary ABC occurring concurrently with a secondary one in the same patient. Although definitive conclusions cannot be based on a single case report, our case seems to show that the pathogenesis could be different for the primary and secondary ABCs. Further reporting of similar cases with multiple mixed lesions could help us in better understanding this disease entity.

   References Top

1.Jaffe HL, Lichtenstein L. Solitary unicameral bone cyst with emphasis on the roentgen picture, the pathologic appearances and the pathogenesis. Arch surg 1942;44:1004-25.  Back to cited text no. 1
2.Bernier JL, Bhaskar SN. Aneurysmal bone cysts of the mandible. Oral Surg Oral Med Oral Pathol 1958;11:1018-28.  Back to cited text no. 2
3.Schajowicz F. Tumour like lesions. In: Schajowicz F. Histological Typing of Bone Tumours. 2nd ed. Berlin: Springer Verlag; 1993. pp 36-42  Back to cited text no. 3
4.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. 4
5.Martinez V, Sissons HA. Aneurysmal bone cyst. A review of 123 cases including primary lesions and those secondary to other bone pathology. Cancer 1988;61:2291-304.  Back to cited text no. 5
6.Tehranzadeh J, Jenkins JJ 3 rd , Horton JA. Case report 249. Osteoblastoma with secondary aneurysmal bone cyst of the frontal bone. Skeletal Radiol 1983;10:276-80.  Back to cited text no. 6
7.Bertoni F, Bacchini P, Capanna R, Ruggieri P, Biagini R, Ferruzzi A, et al. Solid variant of aneurysmal bone cyst. Cancer 1993;71:729-34.  Back to cited text no. 7
8.Kershisnik M, Batsakis JG. Aneurysmal bone cysts of the jaws. Ann Otol Rhinol Laryngol 1994;103:164-5.  Back to cited text no. 8
9.Boyd RC. Aneurysmal bone cysts of the jaws. Br J Oral Surg 1979;16:248-53.  Back to cited text no. 9
10.Struthers PJ, Shear M. Aneurysmal bone cyst of the jaws (II). Pathogenesis. Int J Oral Surg 1984;13:92-100.  Back to cited text no. 10
11.Hillerup S, Hjørting-Hansen E. Aneurysmal bone cyst: Simple bone cyst, two aspects of the same pathologic entity? Int J Oral Surg 1978;7:16-22.  Back to cited text no. 11
12.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. 12
13.Dal Cin P, Kozakewich HP, Goumnerova L, Mankin HJ, Rosenberg AE, Fletcher JA. Variant translocations involving 16q22 and 17p13 in solid variant and extraosseous forms of aneurysmal bone cyst. Genes Chromosomes Cancer 2000;28:233-4.  Back to cited text no. 13
14.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. 14
15.Kransdorf MJ, Sweet DE. Aneurysmal bone cyst: Concept, controversy, clinical presentation and imaging. AJR Am J Roentgenol 1995;164:573-80.  Back to cited text no. 15
16.Sun ZJ, Zhao YF, Yang RL, Zwahlen RA. Aneurysmal bone cysts of jaws: Analysis of 17 cases. J Oral Maxillofac Surg 2010; 68:2122-28.  Back to cited text no. 16
17.Vergel De Dios AM, Bond JR, Shives TC, McLeod RA, Unni KK. Aneurysmal bone cyst. A clinicopathologic study of 238 cases. Cancer 1992;69:2921-31.  Back to cited text no. 17

This article has been cited by
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