Case Description. The authors describe a 10-year-old girl with an expansile lesion of the mandible. The panoramic radiograph showed a well-circumscribed mixed radiolucent-radiopaque lesion of the left mandibular body. An incisional biopsy of the lesion was performed to establish a histologic diagnosis. The specimen was submitted for frozen-section examination, and a diagnosis of CGCG was made. Serum calcium, parathyroid hormone and phosphorous levels were normal. The patient was treated successfully with intralesional corticosteroid injections.

Clinical Implications. Central giant cell lesions have been treated surgically with aggressive curettage. More aggressive and recurrent lesions require resection, which leads to major defects of the jaws. This form of surgical treatment can be particularly disfiguring for a child or young adult. An alternative nonsurgical approach is the intralesional administration of corticosteroids, which have been effective in the treatment of CGCG. If a dentist suspects a patient may have CGCG, he or she should refer the patient to an oral surgeon for follow-up.

This terminology conveyed the idea that the lesion was not a neoplasm. Within the last several years, the word "reparative" has been dropped by most oral and maxillofacial pathologists because the lesion is typically destructive and aggressive, never reparative. Currently, these lesions are designated as giant-cell granuloma or the more noncommittal term "giant-cell lesion."³ The true nature of CGCG remains unknown, despite considerable controversy and discussion in the literature. Many theories have been proposed to explain the etiology and pathogenesis of CGCG. Authorities have viewed it as a reparative process,⁴ a neoplasm of bone⁵ and an anomaly closely related to the aneurysmal bone cyst⁶ but the first two descriptions are now generally regarded to be inaccurate.

CGCG is an uncommon lesion, accounting for less than 7 percent of all benign jaw lesions. The lesion is found predominantly in children and young adults, with more than 60 percent of all cases occurring before the age of 30 years. There is a distinct sex predilection, with a female-to-male ratio of 2:1.⁷ Lesions occur more frequently in the mandible than in the maxilla. Lesions are more common in the anterior region of the jaws, and mandibular lesions frequently extend across the midline.

CGCG usually is an asymptomatic lesion, which may become evident during routine radiographic examination or as a result of painless but visible expansion of the affected jaw. Cortical bone plates are thinned, but perforation into surrounding soft tissue is rare. Radiographically, central giant-cell lesions present as radiolucent defects, which may be unilocular or multilocular.⁸ The defect usually is well-circumscribed and, in some cases, displacement of teeth can be found. The radiographic findings are not specifically diagnostic. Small unilocular lesions can be confused with periapical cysts, and multilocular giant-cell lesions cannot be distinguished radiographically from ameloblastomas or other multilocular lesions.^3,7

Central giant-cell lesions of the jaws are categorized on the basis of radiographic and clinical features.

Central giant-cell lesions of the jaws are categorized on the basis of radiographic and clinical features. Aggressive and nonaggressive lesions have been described, with the likelihood of recurrence a feature of aggressive lesions.^3,7 In addition, the aggressive lesions are found in younger patients, grow quickly, cause pain and induce root resorption and bone perforation.⁹ Studies have failed to identify any biochemical or histologic differences between the aggressive and nonaggressive variants.^10,11 Most studies have looked for differences in giant cells to make such determinations, but no such differences have been found.^12,13

Giant-cell lesions of the jaw exhibit a variety of histopathologic features. Common to all lesions is the presence of multinucleated giant cells in a background of collagenous stroma containing spindle cells. The giant cells are frequently aggregated around numerous vascular channels within the lesion.⁷ Some lesions exhibit considerable fibrosis of the stroma and foci of osteoid and newly formed bone. A patchy distribution of cellular elements is one feature that helps differentiate CGCG from true giant-cell tumors, which are more homogenous. The histopathologic findings closely resemble, and may be identical with, those seen in cherubism, the aneurysmal bone cyst and the brown tumor of hyperparathyroidism.

Surgical management of the CGCG with aggressive curettage is regarded as the treatment of choice.⁹ Resection is performed for recurrent or more aggressive variants, which leads to major defects and loss of teeth. This is particularly mutilating in a growing child or young adult. In such cases, extensive reconstructive procedures are required for anatomic restoration and rehabilitation to achieve satisfactory form and function.

An alternative, nonsurgical approach, first described by Jacoway and colleagues,¹⁴ is the intralesional administration of corticosteroids. Some investigators subsequently reported successful treatment of CGCG with intralesional steroid injections.^15–18 This case report describes the use of intralesional corticosteroid injections to successfully treat a CGCG in the mandible.

	CASE REPORT

TOP ABSTRACT CASE REPORT DISCUSSION CONCLUSION REFERENCES

 
In February 1999, a 10-year-old girl visited her dentist (K.P.) for evaluation of a lesion in the left mandible. The patient’s mother reported that tooth no. 19 had been extracted in June 1998. The original presentation of the lesion at the time of the extraction was that of an exophytic mass surrounding the buccal aspect of tooth no. 19. The results of the biopsy performed at that time showed a peripheral giant-cell granuloma. The patient’s mother reported that the lesion had increased in size since the tooth was extracted.

On physical examination, we found a bony, hard swelling in the patient’s left cheek. Intra-orally, a 3- x 2-centimeter hard buccal expansion was noted in the area of tooth no. 19. The overlying mucosa was intact and no gingival mass was present. Tooth no. 14 was supererupted. No thrill or bruit was noted. We found no neurosensory deficits or lymphadenopathy. Panoramic radiographs showed a 3- x 3-cm well-circumscribed mixed radiolucent-radiopaque lesion of the left mandibular body, extending from tooth no. 18 to tooth no. 20 (Figure 1).

View larger version (119K): [in this window] [in a new window]   Figure 1. Well-circumscribed 3- x 3-centimeter osteolytic lesion in the left body of the mandible.

We decided to administer intralesional corticosteroid injections, following the protocol outlined by Terry and Jacoway.¹⁵ The injections were given on a weekly basis for six weeks. A mixture of 7 cm³ of triamcinolone acetonide and 0.5 percent marcaine with 1/200,000 epinephrine (mixed in a 1:1 ratio) was injected intralesionally on the first and second appointments. On the third appointment, 6 cm³ of the mixture was injected into the lesion. An increase in back pressure was noted on injection. At the fourth through sixth appointments, 6 cm³ of the corticosteroid mixture was injected into the lesion. During the last three treatments, we found it increasingly difficult to perforate the cortical bone for injections. An increase in the amount of back pressure in the syringe was noted during these treatments as well.

Panoramic radiographs obtained immediately after the sixth treatment showed early bone fill of the lesion (Figure 2). Subsequent radiographic evaluations at three and seven months after treatment continued to show good bone consolidation (Figure 3). A decrease in the buccal expansion was first noted at three months. By seven months, the patient’s facial appearance had returned to normal, and we noted almost complete filling of the osteolytic area with new bone (Figure 4). Intraorally, some buccal expansion was still present.

View larger version (113K): [in this window] [in a new window]   Figure 2. Panoramic radiograph taken immediately after six weeks of intralesional corticosteroid injections demonstrates early bone fill of the lesion.

View larger version (119K): [in this window] [in a new window]   Figure 3. Panoramic radiograph taken three months after treatment ended. Notice the bone regeneration within the lesion.

View larger version (117K): [in this window] [in a new window]   Figure 4. Panoramic radiograph taken seven months after the completion of treatment. Notice that the lesion is almost completely filled with new bone.

	DISCUSSION

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Management of CGCG traditionally has been accomplished via surgical removal of the lesion. Some authors recommend en bloc resection including uninvolved bone; others, however, prefer conservative surgical treatment via simple curettage or curettage with peripheral ostectomy,⁹ because these lesions lack characteristics of malignant tumors. Resection of CGCG results in a major defect to the patient. This is of great concern, especially in children and young adults with developing dentition and jaws. Surgical treatment becomes even more difficult in patients with multiple lesions. In such cases, surgery may lead to extensive resection. Radiotherapy has not proven to be a satisfactory alternative, because irradiation of giant-cell lesions may provoke malignant degradation.²⁰

For these reasons, Jacoway and colleagues¹⁴ developed a nonsurgical approach, performed on an outpatient basis, that results in resolution or at least reduction in size of the lesion and allows conservative removal with preservation of adjacent structures. In a 1994 report, Terry and Jacoway¹⁵ confirmed the results of their original abstract.¹⁴ This therapy is an attractive option, particularly for children and young adults.

Because of the usually expansive growth of the CGCG, a thin needle can easily perforate the thin bony cortex overlying the lesion. Intralesional injection of the corticosteroid is preferable to systemic administration to achieve an elevated concentration of the medication in the tissue.

The microscopic similarities between CGCG and sarcoidosis suggest that similar therapeutic regimens would be of value in treating both conditions.^21–23 This perception has been supported by reports showing that intralesional steroid injections into bone cysts resulted in growth of fibrous connective tissue and reossification within three years.²⁴ Similar use of glucocorticoids in aneurysmal bone cysts and nonossifying fibromas was less encouraging.²⁵ Studies by Flanagan and colleagues²⁶ indicated that multinucleated giant cells in giant-cell granulomas of the jaws are osteoclasts, and dexamethasone’s inhibition of osteoclastlike cells in marrow cultures²⁷ supports the use of intralesional corticosteroids for the treatment of CGCGs.

Confirmation of the lesion via biopsy is mandatory before the dental professional can initiate administration of intralesional corticosteroid injections. Caution must be exercised with incisional biopsies, because the giant-cell lesion in bone has many clinical and radiographic similarities to a vascular lesion. A careful medical history, auscultation, palpation and aspiration of the lesion before biopsy may prevent the problem of inadvertently entering a vascular lesion (with the possibility of uncontrollable hemorrhaging). The clinician must perform a careful pretreatment evaluation in all patients to prevent any untoward side effects of therapy. Patients with diabetes mellitus, peptic ulcer, infection and immunocompromised conditions most likely will not benefit from this treatment for obvious reasons.

	CONCLUSION

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Intralesional corticosteroid injections are a good alternative to surgery in the treatment of CGCGs. The technique is simple and inexpensive and allows clinicians to spare vital structures, thus avoiding large defects of the jaws. We are surprised that this simple method of treating a potentially aggressive lesion has not found wide application. This approach, as outlined by Terry and Jacoway,¹⁵ appears to be successful in the treatment of central giant-cell lesions. We believe that sufficient evidence exists to warrant this nonsurgical therapy as the first choice in managing giant-cell lesions in bone. Statistical evaluation of a large number of patients in a long-term prospective study is required to provide documentation and verification of the efficacy of this attractive nonsurgical modality.

View larger version (138K): [in this window] [in a new window]   Dr. Adornato is the chief resident, Department of Oral and Maxillofacial Surgery, Nassau County Medical Center, 2201 Hempstead Turnpike, East Meadow, N.Y. 11554, e-mail "MAdornato{at}AOL.com". Address reprint requests to Dr. Adornato.

View larger version (134K): [in this window] [in a new window]   Dr. Paticoff is an attending staff member, Department of Oral and Maxillofacial Surgery, Nassau County Medical Center, East Meadow, N.Y., and is in private practice, specializing in oral and maxillofacial surgery, Flushing, N.Y.