Volume 3, Issue 4 , Pages 170-173, December 2008
Unusual presentation of multiple giant cell reparative granulomas of the mandible
Article Outline
Summary
Introduction Giant cell reparative granuloma (GCRG) of the mandible is a common benign entity that presents as a single interosseous lesion. It can vary from an asymptomatic slow growing mass to a locally destructive aggressive lesion. Pathological findings of the mandible associated with cherubism and hyperparathyroidism can mimic that of GCRG. However, these diseases demonstrate multiple mandibular lesions as opposed to a single lesion seen in GCRG. We describe a case report of a 9-year-old female with multiple mandibular lesions pathologically positive for GCRG who has no familial history of cherubism or any evidence of hyperparathyroidism. Methods/Results This is a chart review case presentation of a 9-year-old patient who initially presented to the clinic after sustaining trauma to the left side of her face and developed persistant swelling and pain. Imaging showed multiple cystic lesions of the mandiblular rami bilaterally. She underwent curettage and excisional biopsies of these lesions with bone graft. The pathology is consistent with giant cell reparative granuloma. Conclusion This is an unusual presentation of a patient with multiple giant cell reparative granulomas of the mandible. Although this is usually associated with cherubism or hyperparathyroidism, our patient has neither of these diseases.
Keywords: Giant cell reparative granuloma, Cherubism, Hyperparathyroidism, Mandible
1. Introduction
Giant cell reparative granuloma (GCRG) of the mandible is a common benign entity that presents as a single intraosseous lesion. It can vary from an asymptomatic slow growing mass to a locally destructive and aggressive lesion. Pathological findings of the mandible associated with cherubism and hyperparathyroidism can mimic that of GCRG; however, these diseases demonstrate multiple mandibular lesions as opposed to a single lesion seen in GCRG. We describe a case report of a 9-year-old female with multiple mandibular lesions pathologically positive for GCRG who has no familial history of cherubism or any evidence of hyperparathyroidism. We believe that giant cell reparative granulomas should be considered in the differential diagnosis of multiple intraosseous mandibular lesions.
2. Case presentation
Patient is a pleasant otherwise healthy 9-year-old female who was struck on the left side of the face 2 months prior to initial evaluation. There was swelling and pain over the left side of the face that persisted for the duration of the 2 months. She has no difficulty chewing, no change in her occlusion, and no change in her voice. The area has not expanded since the initial swelling was noticed. Her family history was negative for cherubism or any other significant medical conditions. She has no previous medical history or surgical history.
On exam a fairly palpable mass along the left mandible is noted. This mass is extending from the lateral cortex towards the masseter muscle. Mass is non-fluctuant and bony on palpation. Dentition is intact. Occlusion is class I bilaterally. A panorex and CT scan were obtained by an outside otolaryngologist which showed multiple cystic lesions of the mandibular rami bilaterally (Fig. 1).
Fig. 1.
Pre-operative axial cuts of the CT scan of the patient demonstrating cystic lesions at both angles of the mandible (arrows).
She underwent excisional biopsy of the mandibular cyst. The pathology revealed giant cell reparative granuloma (Fig. 2). Further work up revealed no evidence of hyperparathyroidism. She then underwent bilateral excision of mandibular cysts with iliac crest bone graft to repair the defect. She is doing well after the operation and continues to have normal occlusion and normal function. Follow up mandible series and panorex show normal healing (Fig. 3).
Fig. 2.
Pathological slides of the lesion at low (A) medium (B) and high (C) magnification demonstrating multiple giant cells (arrows) in a bed of fibrous tissue and surrounding areas of hemorrhage (*).
Fig. 3.
Post-operative mandible series showing iliac bone graft in the areas of previous cystic lesions (arrows).
3. Discussion
Central giant cell reparative granuloma (GCRG) is an entity first described pathologically by Jaffe in 1953. It is typically a benign lesion of the mandible and maxilla that accounts for about 7% of benign tumors of the jaw bones [1], [2], [3]. The histological features of GCRG as described by the World Health Organization include cellular fibrous tissue that contain multiple foci of hemorrhage, aggregations of multinucleated giant cells and occasionally, trabeculae of woven bone [1], [3]. This lesion occurs more commonly in the mandible compared to the maxilla. There appears to be a propensity to affect women more than men. GCRG is typically described as a singular lesion that is mostly confined to the tooth bearing areas of the mandible [1], [2], [3], [4], [5], [6].
Radiographically these lesions could demonstrate unilocular or multilocular lucency with well-defined or ill-defined borders [1]. The treatment of choice at this point is surgical excision either by curettage or excision of the region of the affected bone. However the recurrence rate of these treatments has been describe as 1–49% in the literature [1], [2], [3]. Other treatments, including calcitonin therapy or intralesional steroid and interferon injections, have been described with variable success. Although calcitonin therapy is promising, it requires years of therapy prior to resolution of the lesion. Intralesional injection with steroids or interferon have had mixed results in multiple trials and are not currently the mainstay of treatment [4], [5].
Multiple lesions of the mandible with similar histological characteristics as GCRG are mostly consistent with cherubism and/or hyperparathyroidism [1], [2], [3], [4], [5], [6]. Cherubism is a disease characterized mainly by bilateral multiloculated cystic expansion of the mandible or the maxilla, which is usually noted at a young age and progresses through puberty. It shows a strong familial inheritance pattern with variable penetrance. Patients classically present with expansion of the mandible or maxilla, full cheeks, and upward casting of the eyes. Pathologically, lesions are consistent with giant cells within a bed of fibrous stroma. Lesions tend to regress through middle age [7]. When multiple giant cell granulomas of the mandible are associated with hyperparathyroidism the disease is known as osteitis fibrosa cystica or brown tumor of the mandible [8]. Our patient had a negative workup for both of these diseases. Our search of the literature did not show any description of multiple GCRG of the mandible without a history of cherubism, hyperparathyroidism, or aneurysmal bone cyst. We believe giant cell reparative granuloma is an independent entity and should be considered as part of the differential diagnosis for multiple intraosseous mandibular lesions.
Acknowledgment
No financial support or funding obtained. Nothing to disclose.
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PII: S1871-4048(08)00025-7
doi:10.1016/j.pedex.2008.03.005
© 2008 Elsevier Ireland Ltd. All rights reserved.
Volume 3, Issue 4 , Pages 170-173, December 2008
