Multicentric giant cell reparative granuloma with extragnathic involvement: Panoramic and computed tomography

Arjit Agarwal; Arvind Shukla; Shruti Chandak

doi:10.4103/2321-3841.151643

CASE REPORT

Year : 2015 | Volume : 3 | Issue : 1 | Page : 15-18

Multicentric giant cell reparative granuloma with extragnathic involvement: Panoramic and computed tomography

Arjit Agarwal, Arvind Shukla, Shruti Chandak
Department of Radiology, Teerthanker Mahaveer Medical College and Research Centre, TMU, Moradabad, Uttar Pradesh, India

Date of Web Publication

18-Feb-2015

Correspondence Address:
Dr. Arjit Agarwal
A-46, Gandhi Nagar, Moradabad, Uttar Pradesh
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/2321-3841.151643

Abstract

Giant cell reparative granuloma is also known as central giant cell granuloma with predilection for the mandible. It is a benign bone tumor, which creates a diagnostic dilemma for radiologist due to their rare occurrence and sometimes aggressive nature. We report a case of young male presenting with a fast growing, large right sided mandibular lump that was bony hard on palpation. There was nasal obstruction along with neck pain. The case was sent for panoramic radiography that revealed multiple radiolucent lesions involving the mandible and the maxillary bone. Further evaluation was done using cross-sectional imaging modality, that is, computed tomography that lead to an elaborate involvement of the mandible and maxilla with incidental lytic involvement of the hyoid bone as well as the C2 vertebral body. Tissue diagnosis was done where diagnosis of giant cell reparative granulomas was confirmed.

Keywords: Giant cell, hyoid bone, mandible, reparative

How to cite this article:
Agarwal A, Shukla A, Chandak S. Multicentric giant cell reparative granuloma with extragnathic involvement: Panoramic and computed tomography. J Oral Maxillofac Radiol 2015;3:15-8

How to cite this URL:
Agarwal A, Shukla A, Chandak S. Multicentric giant cell reparative granuloma with extragnathic involvement: Panoramic and computed tomography. J Oral Maxillofac Radiol [serial online] 2015 [cited 2023 Apr 2];3:15-8. Available from: https://www.joomr.org/text.asp?2015/3/1/15/151643

Introduction

Giant cell granuloma is a rare tumor first described by Jaffe in 1953. ^[1] commonly involving mandible and maxilla, ^[2],[3] however; rare cases have been reported ^[4],[5] in the cranial vault, ^[6] paranasal sinuses, ^[7],[8] temporal bone ^[9] and orbits. ^[10],[11] We found a case of giant cell granuloma involving the mandible, maxilla, hyoid and C2 vertebra simultaneously in an individual that describes the multicentricity of this entity. Literature search reveals a single case in hyoid ^[12] with no case study reporting its occurrence in the vertebral body. The following discussion reviews the literature and presents the case of multicentric giant cell granuloma.

Case Report

A 25-years male presented to the department of otorhinolaryngology with a large swelling in right mandibular region, which increased progressively over the period of 2 years with rapid growth in recent 2 months. There was associated nasal obstruction with neck pain. He was a nonsmoker, with no previous history of trauma and radiation exposure.

On physical examination, the lump was bony hard with a palpable mass in the nasal region causing nasal block bilaterally. Associated loss of multiple teeth in the lower jaw was there on the same side. There were no palpable lymph nodes in the neck. Routine biochemical test results including serum calcium (9 mg/dl), alkaline phosphatase (50 IU/L), phosphorus (2.5 mg/dl), renal function (serum creatinine-0.8 mg/dl; urea-20 mg/dl), and parathyroid hormone (25 pg/ml) were within normal limits.

The patient was sent for panoramic radiography [Figure 1], which revealed a large expansile mandibular lesion involving the right ramus and condyle along with similar aggressive lesion in the left maxillary and nasal region causing bony destruction. Computed tomography was done with multiplanar reconstruction [Figure 2] and volume rendering [Figure 3], which gave the elaborate involvement of the mandible and thinning of both buccal as well as the lingual cortices of the mandible and maxilla with intralesional osseous elements because of destruction and bony remodeling. There was incidental visualization of the lytic lesion in the left side of the body of the hyoid bone and the body of axis vertebra [Figure 4]. Associated concentric height reduction of the C5 vertebral body was also noted.

Figure 1: Panoramic radiograph revealing loss of multiple teeth with expansile right mandibular lesion in ramus and condyle (solid arrow). Lytic lesion in left maxilla with bone destruction (open arrow)

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Figure 2: (a) Coronal computed tomography (CT) in bone window showing right sided mandibular expansile lytic lesion with bone remodeling (solid arrow). Lytic lesion in left side of the body of hyoid bone (open arrow), (b) axial CT in soft tissue window showing lytic destructive lesion in left maxilla with nasal obstruction (solid arrow)

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Figure 3: (a) Computed tomography (CT) left parasagittal section showing lytic lesions in left maxilla and hyoid bone (solid arrow), (b) CT mid-sagittal section showing lytic destruction of the axis (C2) body (solid arrow)

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Figure 4: (a) Volume rendered three-dimensional computed tomography (CT) coronal image showing pedunculated lytic lesion in the right mandibular ramus (solid arrow), (b) volume rendered three-dimensional CT sagittal section showing lytic lesion involving hyoid bone (solid arrow)

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The lesions arising from the mandible and maxilla were biopsied. The patient underwent surgical resection for the mandibular lesion, and right hemimandibulectomy was done with en bloc resection of the mass and mandibular reconstruction. Histopathological examination from both the biopsied as well as the resected samples revealed fibroblastic proliferation with background of mononuclear cells and interspersed multinucleated giant cell [Figure 5].

Figure 5: Micrograph of the tissue in H and E

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Discussion

Giant cell reparative granuloma is a benign tumor commonly involving mandible and maxilla usually occurring in the first two decades of life. ^[5],[13] They are twice more common in females than males. ^[6] To our knowledge, this is the first case of giant cell granuloma involving multiple bones viz., maxilla, hyoid and vertebral body apart from its common affinity toward mandible.

These tumors need to be differentiated from other craniofacial lytic bone lesions like giant cell tumor, cherubism and brown tumor of hyperparathyroidism. In our case, there was no significant mitotic activity with few giant cells and hemorrhagic areas which are not in favor of giant cell tumor. Further biochemical parameters were within normal limits with no familial history ruling out both brown tumors and cherubism, i.e., familial fibrous dysplasia of the jaw. Some authors consider both central giant cell granulomas and giant cell tumors as a continuum of the same disease process with later being aggressive with predilection for long bones. ^[14],[15]

There are two clinical forms of central giant cell granulomas which are:

Central-endosteal and
Peripheral soft tissue. ^[16] The later form is more common in the mucosal region of the gingiva and alveolus with predilection for females of <30 years of age.

Endosteal variety is common in the mandible in the age group of 10-20 years, however; histopathologically both the varieties cannot be differentiated. ^[17] Central-endosteal form is further classified into nonaggressive and aggressive subtypes according to the destruction and bony remodeling. Hence in this case, we came across the central-endosteal form of the tumor with aggressive subtype. Aggressive nature is ascertained by clinical and radiological features that included rapid growth, increased pain, large size of the lesions in mandible and maxilla (>5 cm) and bony destruction (cortical thinning). ^[15]

Pathogenesis of the entity is still dubious and its origin is ascertained to be either hamartomatous, reactive or neoplastic where most of the authors feel that it is sequel to posttraumatic osseous hemorrhage and periosteal reaction, ^[1],[6] though in our case there was no such precedent event. Many studies have implicated their genetic origin. ^[18],[19] Giant cell reparative granuloma is characterized by vascular stroma composed of oval and spindle-shaped fibroblastic cells with areas of hemorrhage, abundant hemosiderin and fibrosis. The tumor has been branded as "reparative" by Worth where the author has reported a series of cases of the entity, which were not treated surgically and healed spontaneously, ^[20] however; these lesions show inconsistent behavior and hence better known as central giant cell granulomas.

Radiological diagnosis of these lesions is difficult because of their uncommon occurrence. There are many histopathological tools which aid in confirmation of the entity apart from routine staining procedures which include immunohistochemistry of the lesions using tartrate resistant acid phosphatase which demonstrates uniform reactivity in the cytoplasm of the polynuclear-giant cells with minimal reactivity in mononuclear cells. Similar affinity with CD-68 is seen where moderate reactivity is also demonstrated by the mononuclear cells. Ki-67 staining is specific to the mononuclear cells with absent reactivity to multinucleate giant cells. Multicentric presentation of the central giant cell granulomas is still rare, which adds to the diagnostic dilemma for the radiologist. Involvement of the hyoid bone and vertebral body alerts us regarding the complete skeletal evaluation of any mandibular lesion suspicious of giant cell reparative granuloma.

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