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2021, Number 4

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Rev Odont Mex 2021; 25 (4)

Aggressive central giant cell granuloma of the maxilla: treatment with triamcinolone infiltration and curettage

Nieto Munguía, Ana María1; Mendoza García, Raúl2; González Rebattú y González, Mauricio3; Cázarez Ríos, Víctor Daniel4

Language: English/Spanish [Versión en español]
References: 20
Page: 368-375
PDF size: 268.86 Kb.


ABSTRACT

Introduction: central giant cell granuloma (CGCG) is a locally aggressive, benign osteolytic lesion of the jaws characterized by the presence of osteoclast-like giant cells in a vascular stroma. About 30% of cases present aggressively, characterized by rapid growth, greater than 5 cm, pain, cortical perforation and invasion of peripheral tissue and root resorption. They are aggressive and non-aggressive. Objective: to present the case of an aggressive central giant cell granuloma treated with several therapeutic resources. Case presentation: a 35-year-old female patient with an aggressive central giant cell granuloma on the right side of the maxilla, treated with triamcinolone infiltration following the Jacoway protocol and subsequent enucleation and surgical curettage. Results: the total size of the lesion was reduced by approximately 30%, followed by enucleation and curettage, with a year control; currently no recurrence. The use of auxiliary therapies to surgery, such as intralesional corticosteroid infiltration, is a useful treatment in the management of central giant cell granuloma, allowing surgery to be less radical and traumatic in most cases, therefore, and a decrease in morbidity in the patient.



INTRODUCTION

The World Health Organization defines central giant cell granuloma (CGCG) as a benign, locally aggressive osteolytic lesion of the jaws, characterized by the presence of osteoclast-like giant cells in a vascular stroma. It represents 10% of all benign tumors of the jaws. These lesions are more frequent in the anterior region of the jaw and the presence of multiple lesions are related to LEOPARD, Noonan or neurofibromatosis type 1, syndromes.1 The etiology of CGCG is uncertain, they describe it as an inflammatory proliferation; it can also behave as an aggressive neoplasia process.2 Jaffe regards this tumor as a locally reparative bone reaction due to inflammation, local trauma, or hemorrhage.3 Worth attempted to demonstrate this theory using radiographic evidence, showing bone repair of untreated lesions.4

The most frequent characteristics of CGCG are slow growth, asymptomatic, with cortical expansion without perforation or involvement of the adjacent teeth. According to Choung, around 30% of cases present aggressively, characterized by rapid growth, size greater than 5 cm, pain, perforation of the cortex and invasion of the peripheral tissue and root resorption; these have a high recurrence rate after surgical curettage.5 The multinucleated giant cells are osteoclast type with antigenic properties and phenotypic markers of mononuclear precursors.6 The literature reports a high recurrence rate for surgical curettage, especially in aggressive lesions.7-9 Steroids are believed to inhibit giant cells through lysosomal protease, preventing bone resorption; also steroids induce apoptosis of osteoblast-like cells.10,11

Calcitonin has been used successfully to treat these lesions. Its use is based on the histological resemblance of CGCG to the brown tumor of hyperparathyroidism. It has antagonist properties with parathormone. Therefore, giant cells are thought to have receptors for calcitonin.12 On the other hand, treatment with alpha interferon is based on the suppression of growth factors, which could reduce angiogenesis. Therapy consists of subcutaneous administration and monitoring of the fibroblast growth factor in the urine. Some protocols suggest starting with surgical treatment, followed by at least six months of alpha interferon therapy.13 Inhibitators of the receptor activator for nuclear factor kappa-β have been developed as an effective alternative for the treatment of this type of lesions, mainly in the aggressive type, since they inhibit osteoclasts; however, studies about the jaws have not yet been significant.14

The intralesional infiltration of steroids in CGCG was reported for the first time by Jacoway et al. in 1988.15 In 1994, Terry and Jacoway16 formulated the treatment protocol by means of a weekly injection of a mixture of equal parts triamcinolone acetonide (10 mg/mL) and a local anesthetic (0.5% bupivacaine with epinephrine 1: 200,000). The recommended dose is 2 mL/2 cm radiolucent zone and applying the injection in different areas of the lesion for six weeks at least. Steroid treatment is based on the microscopic resemblance between CGCG and sarcoidosis. Supported by a study with dexamethasone on osteoclasts, Hirayama states that these drugs have a direct effect on the formation and activity of these cells and inhibit the activity of mature osteoclasts.17



CASE REPORT

A 35-year-old female patient, who in June 2017 presented an asymptomatic increase in volume in the right genian region, approximately four months old.

On clinical examination, a slight facial asymmetry was appreciated at the expense of an increase in volume in the right nasolabial region of approximately 4.0 × 4.0 centimeters, without alterations in the skin. Intraorally, a loss of the fundus of the mucogingival fold was observed due to the increase in volume that goes from the area near the maxillary triangular process to the alveolar border near the first premolar on the right side. Well defined, smooth surface, same color as adjacent mucosa, firm consistency, asymptomatic on palpation, with extrusion of the first and second premolars. A simple computerized axial tomography shows a mixed lesion: hyperintense and hypodense areas with soft tissue occupying the maxilla on the right side. It extends through the maxillary body, the pyramidal process, a large part of the maxillary sinus and the alveolar region from the canine to the first ipsilateral molar, with displacement of the same, expansion and perforation of the vestibular cortex (Figure 1). According to clinical and imaging characteristics, the presumptive diagnosis corresponds to CGCG. Incisional biopsy is indicated, compatible with CGCG; to rule out brown tumor of hyperparathyroidism, parathormone levels are requested, which are at standard levels.

The intralesional infiltration protocol was started based on the protocol described by Jacoway. 3.5 mL of triamcinolone acetonide was injected with 3.5 mL of 2% lidocaine with 1:100,000 epinephrine once a week, until completing six applications. The tumor decreased discreetly (Figure 2), so enucleation and curettage of the lesion were performed under balanced general anesthesia; the specimen was completely extracted and then it was 10% formalin-fixed (Figure 3).

A histopathological study was done, which confirmed the diagnosis of central giant cell granuloma (CGCG). A year later, a simple control computed tomography was performed which showed a favorable evolution, no data of recurrence, and the maxillary bone in clear recovery. Currently the patient is progressing adequately without associated symptoms (Figure 4).



DISCUSSION

Dolanmaz18 published the treatment of seven CGCG patients managed with intralesional infiltration with 3.5 mL of triamcinolone acetonide (Kenacort-A) and 3.5 mL of 0.5% bupivacaine with epinephrine at 1:200,000, weekly for a six weeks lapse, following Terry and Jacoway's protocol. No side effects are reported in any patient due to the use of steroids. Four patients showed complete resolution and ossification. The remaining patients did not present absolute resolution but did show a considerable decrease in size of the lesion; after two years control, they reported no recurrences. The size of our patient's lesion was slightly reduced.

In 2012, Rachmiel et al.19 reported the treatment applied to a 24-year-old female patient with aggressive CGCG, using a scheme of intralesional injections of Kenacort-A with 2% lidocaine with 1:100,000 epinephrine, in a dose of 1 mL/1 cm radiolucent lesion determined by orthopantomography, during six weeks and calcitonin nasal spray at 200 IU daily for three months; subsequently a surgical curettage with peripheral osteotomy was performed, preserving the continuity of the mandible and dental organs. Reconstruction was performed immediately with an autologous iliac crest graft; the five years control shows no evidence of recurrence. As the previous authors did, we also followed the Jacoway protocol. In our case we did not use calcitonin and the reconstruction was different, since lesion was in the maxillary body.

Nogueira et al.20 report 21 cases of CGCG in the maxilla and mandible treated with intralesional injection of triamcinolone hexacetonide (20 mg/mL) diluted in an anesthetic solution of lidocaine 2% with 1:200,000 epinephrine, in a ratio of 1:1; 1 mL of solution infiltrated for every 1 cm3 radiolucent lesion, with a total of six weekly applications. Ten of these patients had aggressive lesions and 11 had non-aggressive lesions. Two patients showed a negative response and required surgical resection; four showed a moderate response and fifteen with an adequate response. Eight of the 19 who showed a moderate to good response required osteoplasty to restore facial aesthetics. Nogueira's study is very useful in the evaluation of these lesions since they distinguish aggressive and non-aggressive, being similar that the aggressive type responded discreetly, requiring additional surgical treatment.

In our case, the patient presented an aggressive CGCG occupying almost completely the maxillary body on the right side. Because removing the lesion in its entirety only by surgical treatment would risk the patient's health, it was decided to start by infiltrating corticosteroids, as stated by the Jacoway protocol, achieving a clinical reduction of the lesion by 30%. We thought that the aggressiveness of the lesion would not allow its total remission applying only steroid infiltration therapy, coinciding with what has been reported in the literature. However, this allowed the surgical procedure to completely remove the lesion and a year control showed no recurrence, with frank bone regeneration, no data on facial deformity or significant comorbidities.



CONCLUSIONS

The use of auxiliary surgical therapies such as intralesional corticosteroid infiltration is a useful treatment in the management of CGCG, since it significantly helps to reduce the size of the initial lesion, producing a less traumatic and less radical trans-surgical procedure. In this case, the Jacoway protocol was used to apply triamcinolone to the lesion, reducing its size and removing it without complications later, by surgery. Currently, the patient shows an adequate evolution, with formation of bone tissue in the procedure bed, as observed in the one-year follow-up of the computed tomographic study.


REFERENCES

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AFFILIATIONS

1 Cirujana Maxilofacial adscrita al Servicio de Cirugía Maxilofacial del Hospital Regional "Lic. Adolfo López Mateos", ISSSTE. México.

2 Cirujano Maxilofacial, Secretaría de Salud. México.

3 Cirujano Maxilofacial adscrito al Servicio de Cirugía Maxilofacial del Hospital Regional 1o de Octubre, ISSSTE. México.

4 Residente de Cirugía Maxilofacial del Hospital Regional "Lic. Adolfo López Mateos", ISSSTE. México,



CORRESPONDENCE

Ana María Nieto Munguía. E-mail: annie_tit@hotmail.com




Received: Marzo 2020. Accepted: Julio 2020.

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