Journal of Datta Meghe Institute of Medical Sciences University

CASE REPORT
Year
: 2021  |  Volume : 16  |  Issue : 1  |  Page : 199--201

Recurrence of aneurysmal bone cyst of the mandible and its management: A case report and overview


Akash Kasatwar1, Sandeep Patil2, D Durairaj3, Shree Bajaj4,  
1 Department of Dentistry, District General Hospital, Chandrapur, Maharashtra, India
2 Department of Oral and Maxillofacial Surgery, PDU Dental College and Hospital, Solapur, Maharashtra, India
3 Department of Oral and Maxillofacial Surgery, Adiparashakti Dental College and Hospital, Melmaruvathur, Kancheepuram, Tamil Nadu, India
4 Department of Oral Pathology, Sharad Pawar Dental College and Hospital, DMIMS, Sawangi (Meghe), Wardha, Maharashtra, India

Correspondence Address:
Dr. Sandeep Patil
Department of Oral and Maxillofacial, PDU Dental College and Hospital, Solapur, Maharashtra
India

Abstract

Aneurysmal bone cyst (ABC) was first described by Jaffe and Lichtenstein in 1942 characterized by the blow out of the bone seen in radiograph. 12% of the patients had a lesion in the head and neck region, of which only 2% of these pathologies occur in the maxillofacial region. It is most common in those regions of the skeleton where there is a relatively high venous pressure and high marrow content. Mandibular and maxillary molar regions are most commonly involved followed by angle and ascending ramus with preponderance of female patients. In the present case, a 52-year-old female reported to our institute with the chief complaint of swelling. Clinically and correlating radiographically, a large lesion measuring 6 cm × 2.5 cm approximately was involved occupying half of the mandible. This article describes the clinical, radiographical, and histopathological picture, differential diagnosis, and the management of a recurrence of large ABC of the mandible.



How to cite this article:
Kasatwar A, Patil S, Durairaj D, Bajaj S. Recurrence of aneurysmal bone cyst of the mandible and its management: A case report and overview.J Datta Meghe Inst Med Sci Univ 2021;16:199-201


How to cite this URL:
Kasatwar A, Patil S, Durairaj D, Bajaj S. Recurrence of aneurysmal bone cyst of the mandible and its management: A case report and overview. J Datta Meghe Inst Med Sci Univ [serial online] 2021 [cited 2021 Oct 21 ];16:199-201
Available from: http://www.journaldmims.com/text.asp?2021/16/1/199/322635


Full Text



 Introduction



Aneurysmal bone cyst (ABC) is a riveting benign reactive lesion of the bone rather than a true neoplasm. This unique lesion was separated as a distinct entity by Jaffe and Lichtenstein in 1942, and the term ABC for this pathology was given describing the characteristics blow out as seen in radiographs.[1] It is most commonly located in long bones and vertebra and very rarely in the craniomaxillofacial region. The etiology of this lesion is still uncertain and controversial. In the head and neck region, it is more frequently reported in the mandible than the maxilla in the ratio of 3:1, with preponderance for the body, ramus, and angle of the mandible.[2] Often they are mistaken for other common lesions in the maxillofacial region due to their similar clinical and radiological appearance. ABC can be classified into three types. Conventional or vascular type 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 radiolucency on a routine radiograph or as a small swelling. 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.[3] An extensive study was undertaken to test the hypothesis that an ABC is a secondary phenomenon that occurs in a primary lesion of the bone. The purpose of this article is to report a rare case of the recurrence of ABC in a 45-year-old female affecting the body of the mandible and its management.

 Case Report



A 52-year-old female patient reported to hospital with chief complaint of pain in the mandibular right-sided molar region for the past one year. The patient's previous treatment reports suggest that she underwent curettage as a primary surgical treatment for the lesion in the private hospital with histopathological report giving diagnosis as ABC. On examination, slight asymmetry was apparent on the right side of the face. Intraoral examination revealed obliteration of the right side vestibular sulcus in the premolar and molar region. On palpation, a firm swelling measuring about 4 cm × 2.5 cm noticed in the right side body region of the mandible extending from lateral incisor to molar region. Panoramic radiograph revealed unilocular lesion of size about 6 cm × 3 cm with sclerotic margins and resorption of roots of second premolar and first molar [Figure 1]a. Taking into consideration the previous histopathological report, aspiration of the lesion with wide bore needle (18 gauze) was done to rule out hemangioma or arteriovenous malformation. Aspiration showed very less blood-tinged fluid. Aspiration showed very less blood-tinged fluid. Incisional biopsy was then done under local anesthesia, and histopathological diagnosis of recurrence of ABC was confirmed.{Figure 1}

Since the lesion was larger,general anaesthesia was preferred for the surgery. Considering the aggressive nature of the lesion and recurrence,segmental resection of the mandible was done [Figure 1]b and [Figure 1]c. Mandibular anatomy and facial symmetry were maintained with the reconstruction plates. The patient was under the follow-up for 15 months without any signs and symptoms of recurrence.

 Discussion



ABC is basically classified as benign nonodontogenic lesion containing multinucleated giant cells along which also includes giant cell tumor, the giant cell granuloma, hyperparathyroidism, and cherubism [Figure 1]d. ABCs are benign neoformations which occur very rarely in the jaws. The etiology and pathogenesis of the lesion are still unknown and controversial. Various theories have been proposed in the literature regarding the pathogenesis of ABC. First, Lichtenstein has proposed that ABC arises as a result of persistent local alteration in hemodynamics, leading to increased venous pressure and subsequent development of dilated and engorged vascular bed in transformed resorbed bone area. Another alternative explanation is that hematoma maintains a circulatory connection with the damaged vessels that may produce a slow flow of the blood through the lesion and account for the clinical “welling” up of the blood.[4] Biesecker proposed a new hypothesis that a primary lesion of the bone initiates an osseous, arteriovenous fistula and thereby creates secondary reactive lesion of the bone, i.e., ABC. Thus, ABC is a secondary reactive pseudocystic lesion occurring in conjunction with primary pathologies of the bone.[5] These secondary ABCs account for nearly 30 % of the lesion.[6] Levy et al. reported 57 cases of ABCs most commonly associated with unicameral bone cyst, osteoclastoma, and osteosarcoma.[7] Molecular evidence that now exists supports the theory that vascular injury is important in the development of this lesion. In the present case, the patient gives the history of trauma.

ABCs can occur at any age of life but usually occur at the first and second decade of life, and most of the cases are occurring before the age of 30. The average age of occurrence is 13 years and 80% of patients are <20 years old. Majority of the cases tend to have predilection to occur in female patients. ABC is of distinct variety involving all parts of the body skeleton, but majority of the cases are seen the long bones and vertebral column. Other sites most commonly involved are clavicle, rib, innominate bone, skull, and bones of the hand and feet. ABCs occur with very less frequency in the oral and maxillofacial region. Most of the cases are reported in the mandible than the maxilla. Of the reported cases, two-thirds were located in the mandible (the body of the mandible 40%, the ramus 30% and the angle 19%) while one-third in the maxilla. Considering all types of jaw cysts, the ABC is extremely rare with only 0.5% overall occurrence in the oral region.[1] In the present case also, the lesion was involving the mandibular premolar and molar regions that correlate with the other studies in the literature be destruction or perforation of the cortex and a periosteal reaction may be evident. In the present case, unilocular radioluscency was seen in premolar-molar regions with resorption of the roots.

Clinical presentations of the ABC range from mild, slowly expanding, semisolid growth causing slight facial asymmetry to a rapidly expanding vascular swelling causing extensive bone destruction and mimicking malignant lesions.[1] The present case shows that slowly growing lesion and expansion of the bony cortex. The radiological features of ABC in the jaws are quite variable. It may appear radiolucent, radiopaque, or mixed. The bone may be expanded, may appear cystic with honeycomb or soap bubble appearance, and may be eccentrically ballooned.[3] Histologically, the present case of ABC consists of multinucleated irregular-shaped giant cells with blood-filled cystic spaces.

Various treatment methods have been described in the literature including enucleation, curettage, embolization, cryotherapy with curettage, percutaneous sclerotherapy, low-dose radiation, and resection followed by suitable reconstruction.[8],[9],[10] The modality of the treatment depends on the size of the lesion and clinical and radiological aggressiveness of the pathology. To date, surgical curettage or partial resection is the primary choice of the treatment. Percutaneous sclerotherapy can be used for the treatment of ABC, but the studies regarding its use in the jaws is anecdotal. Cryotherapy can be used for a very small lesion but lacks sufficient data to prove the efficacy of this method in the management of ABC. Smaller lesions <1.5 cm can be treated by complete curettage alone. However, there are chances of recurrence if incompletely removed. Studies have shown to have 50% recurrence after curettage in long bones.[5] Some studies reported that ABCs have a high recurrence rate (20%–30%), and they are usually recurring within the 1st year after the initial treatment.[3] Low-dose radiation therapy was also used for the treatment. However, considering the lesion is benign and the possibility of radiation sarcoma precludes this treatment modality.[6] Therefore, surgical resection has been considered as a choice for complete removal of aggressive ABCs. In the present case, considering the aggressive nature and recurrence within 1 year of curettage with <5 mm of bone remaining at lower border, segmental resection of the lesion was done with clear surgical margins. A 2.5 mm reconstruction plate was then fixed to maintain the lower border continuity of the mandible. Routine follow-up was done for a period of 2 years. Surgical site was healthy with no evidence of recurrence in both clinical and radiographic evaluation. Since their original lookout, many cases of ABC have been reported in the literature with various treatment modalities. Treatment of this type of lesion should be according to the site, size, aggressiveness, and recurrence. Surgical excision of the ABC with aggressive approach of either segmental resection or hemimandibulectomy should be done to avoid the recurrence of the pathology.[11],[12],[13],[14],[15]

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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