2021, Number 4
Giant cell central granuloma in a pediatric patient
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ABSTRACT
Introduction: giant cell tumors constitute a set of pathological processes with a certain degree of controversy in diagnosis, different in clinical behavior but with histological characteristics in common. The central granuloma of giant cells (GCCG) is an intraosseous lesion of cellular fibrous tissue with multiple hemorrhagic foci and nucleated giant cell aggregations. The objective of this paper is expose the clinical features observed, approach and following of one girl with giant cell central granuloma. Case report: this is a 6-year-old female patient who was referred to the Maxillofacial Surgery Service of the General Hospital of Zacatecas, with no relevant medical history. The intraoral examination showed a remarkable increase in the volume of sessile appearance and reddish, painful palpation, located between the inner side of the upper labial mucosa and frenulum, and extended to the right canine, causing asymmetry in the anterior portion of the hard palate. After carrying out imaging studies (radiographic and Cone Beam), the surgical removal of the lesion was performed under general anesthesia, and the giant cell granuloma was confirmed histologically. Conclusion: in oral lesions the choice of treatment is influenced by the clinical behavior of the lesion. The age of the patient, the site and the extension are also contributing factors. Early detection in children allows conservative management, resulting in decreased undesirable loss of oral structures.INTRODUCTION
Giant cell lesions are a group of lesions that affect the jaws and have common histopathologic features with variable content.1 Three groups of lesions containing giant cells have been found, the first comprising different processes such as fibrous dysplasia, cherubism, Paget's disease, among others; and a second and third group consisting of central giant cell granuloma and true giant cell tumor, respectively.2
Central giant cell granuloma (GCCG) was first described by Jaffe (1953) as a reparative giant cell granuloma,3 the World Health Organization (WHO) defines it as an intraosseous lesion of fibrous cellular tissue containing multiple foci of hemorrhage, multiple aggregations of nucleated giant cells and sometimes trabeculae of bone tissue,4 it is of controversial etiology, it is suggested that it is a reactive lesion, developmental anomaly or a benign neoplasm. It is the most common, although it is infrequent since it represents only 7% of all benign lesions of the jaws;4 it is preferentially located in the mandible (70%)2,5 in the anterior region, although it is not unusual among premolars and molars, in the maxilla it is mostly located in the anterior section.2
Based on its clinical and radiographic characteristics, two categories have been proposed: a) non-aggressive, asymptomatic, slow-growing, without perforation of bone cortices or root resorption, and low tendency to recurrence; b) aggressive, fast-growing, painful, with paresthesia, expansion and/or perforation of bone cortices, root resorption and high tendency to recurrence (11-49%).5
In the early stages, its detection occurs fortuitously, after the physical or radiographic examination. Its biological behavior may change from non-aggressive to aggressive, with pain, resorption, tooth and/or germ displacement, and rapid growth.4,6,7 In advanced lesions it is possible to observe an evident facial asymmetry, in addition to spontaneous nasal bleeding and nasal obstruction, when there is an invasion of the maxillary sinus space.8 Its radiographic appearance is diverse, such as uni- or multilocular areas that may be associated with adjacent teeth, tooth and/or germ displacement, root resorption, and bone perforations.7-9 The differential diagnosis in small lesions is with periapical granulomas or cysts, and ameloblastomas in multilocular lesions.3 The use of cone-beam computed tomography is useful in the diagnosis of lesions of this type.7 Histopathologically, it contains foci and mantles of multinucleated giant cells of variable shape and size in a fibroblastic stroma composed of ovoid or spindle cells showing a high mitotic index. The vascular density is prominent with marked erythrocyte extravasation and hemosiderin deposits, and the number of nuclei varies from a few to several dozen.10
The aim of the present study is to report the clinical and imaging features, approach, and follow-up of a pediatric patient with central giant cell granuloma.
CASE REPORT
A female patient aged six years two months, referred by a general practice dentist to the Maxillofacial Surgery Service of the General Hospital of Zacatecas "Luz González Cosío", because she presented "a small ball in her mouth" as referred by her mother, six months before in a dental check-up in her place of residence, she had an extraction of the OD 51 because it was associated with a bulge in the gum, which evolved during the following weeks increasing its volume. After the informed consent form had been completed and signed by the responsible person, a medical history was taken, which revealed a patient with no relevant pathological, personal, or family history. The general physical examination showed the good general condition, with normal vital functions and psychomotor development.
Facial examination (Figure 1) showed a clear asymmetry and moderate deformity associated with the upper lip. On intraoral examination, the upper labial mucosa showed increased volume above the labial frenulum, extending towards the right upper canine (Figure 2A) involving the alveolar ridge; it was also observed in the anterior hard palate, with a sessile and reddish appearance, with pain on palpation. Dentally, the patient was in mixed dentition, the absence of OD 51 was due to a previous extraction, OD 61 showed distal displacement (Figure 2B). OD 71 and 81 had physiologically exfoliated, and OD 41 was in early eruption. In the lower arch the dental organs 72, 73, 74, 75, 41, 82, 83, 84, and 85 were observed, the lower deciduous molars showed caries lesions in enamel and dentin, there was no soft tissue alteration in that arch (Figure 2C); and in the upper arch the DOs 52, 53, 54, 55, 62, 63, 64 and 65 were present.
Radiographically, a wide unilocular area was observed (Figure 3), which caused dental and permanent central incisor germ displacement due to its extension towards the floor of the nasal cavity. Cone-beam computed image showed the presence of a 25 mm mass in most of its dimensions (Figure 4).
A cystic lesion was ruled out, and after multidisciplinary evaluation, surgical excision was performed under general anesthesia (Figure 5). Local anesthetic (lidocaine/epinephrine) was infiltrated, followed by incision with scalpel and electroscalpel for subsequent removal (Figure 6). With the aid of a curette, multiple firm yellow fragments were obtained (25.0 × 25.0 × 10.0 mm the largest, and smaller 5.0 × 5.0 × 4.0 mm) and sutured. The presence of multinucleated giant cells was determined histopathologically (Figure 7), which confirmed that it was a central giant cell granuloma. The stitches were removed 10 days after surgery.
The patient was examined three weeks later at the Pediatric Dentistry Clinic of the Autonomous University of Zacatecas to evaluate the clinical and radiographic evolution (Figure 8), where the healing of the soft tissues involved was observed. It was also observed the exposure of a central incisor in the vestibule of the lesion, and radiographically the presence of teeth 11, 21, and 22 was appreciated even with incomplete root formation.
Monthly radiographic control was indicated to follow up on the dental germs present, bone formation and to monitor the possibility of recurrence of the granuloma, to finally consider oral rehabilitation, however, he does not adhere to the follow-up plan and does not attend the control appointments, which could be because he does not live in a place close to the clinic facilities.
DISCUSSION
The treatment of GCCG should be individualized for each case, taking into account the patient's age, general health condition, the development and location of the lesion, as well as its evolution and the degree of penetration into the surrounding tissues.
They can appear at any age, but most cases occur between 10 and 30 years of age,5 in a female to male ratio of 2:1,8 in the case of the patient reported here, the GCCG started at an early age before five years of age, and with aggressive behavior, causing color changes in the mucosa that can go from a bluish or brownish tone;8 which depending on their location and size can cause tooth displacement or mobility resulting in malocclusion and malposition.11
A major problem related to the destructive nature and high recurrence of GCCG derives from the performance of repetitive surgical procedures with questionable benefits, due to the important functional and esthetic sequelae. These are probably of greater significance when the lesion affects the pediatric or adolescent population.5 The classic treatment of GCCG is the excision of the lesion since it allows the totality of the lesion to be eliminated by simple curettage. In aggressive lesions, treatment ranges from curettage of the lesion bed with cryosurgery, surgical resection with peripheral osteotomy and en bloc resection, in which case patients are left with important sequelae such as the loss of tooth germs, adjacent teeth or extensive areas of the jaws.11-14 Currently, coadjuvant and conservative treatments have been reported, such as the use of intralesional corticosteroids, treatment with calcitonin, interferon-alpha and human monoclonal antibodies (RANK-L) and tyrosinase inhibitor proteins such as imatinib.8,11,15
In 2007, Delgado-Azañero et al. report the case of a 9-year-old female patient with a multilocular GCCG located in the mandibular symphysis, with aggressive osteolytic behavior. The presence of GCCG was confirmed through biopsy and histopathological study; and was treated with intralesional injections of triamcinolone and lidocaine, at the end of 15 sessions a favorable evolution was observed by reducing the size of the lesion, accompanied by recovery and remodeling of the bone structure around the teeth and continuity in the formation of the tooth germs associated with it.5 Janas et al., describe the case of a central granuloma located in the maxilla, close to the premolars on the right side, in an 8-year-old boy, which was observed as a lump on the hard palate, asymptomatic, with normal features of the lining mucosa. Radiographically it was described as an osteolytic unilocular lesion of the maxillary bone with a displacement of a tooth germ (OD 14). Aspirational biopsy and histopathological study were performed to confirm the lesion, and the tumor and associated tooth were surgically removed under general anesthesia. After two years of follow-up, no recurrence of the lesion was observed.16
In contrast to Janas (2015) and Delgado-Azañero (2007) the case presented was a 6-year-old patient with a granuloma of unfavorable evolution with involvement of bone structures and tooth germs, which for its excision required extensive structural removal, and with difficulties in performing its dental clinical follow-up. Radical surgical removal of the GCCG has also been reported in the pediatric population, such is the case of the patient-reported by Wang et al., in which en bloc resection of a large and aggressive GCCG was found in a 6-year-old female patient involving the mandibular condyle was performed.17 The efficacy of alternative treatments has been explored, although surgical intervention continues to be the standard of care, mainly when aggressive behavior is observed,9 enucleation, and curettage of the niche are favorable for the preservation of the bone cortex and adjacent nerves, in child patients as was our case, it is important to seek the preservation of the facial structures involved, to obtain the best long-term results.
CONCLUSION
GCCG is a difficult lesion to diagnose, due to the low frequency of its occurrence, its characteristics, and clinical behavior, in addition to the individual characteristics and socioeconomic status of the patient. Early detection in those affected allows conservative management, which results in a decrease in the undesirable loss of oral structures since they are pathologies with undesirable esthetic and functional consequences.
ACKNOWLEDGMENTS
Dra. Bertha Berthaud González (médico anatomopatólogo del Hospital General de Zacatecas "Luz González Cosío"), M.C.D. Omar Geraldo Ramírez Rivera (residente de especialidad en Odontopediatría), M.C.D. Francisco Javier Mejía Pérez, M.C.D. Karen García Herrera,† M.C.D. Carlos Pérez Ponce, M.C.D. Jeremy Alfonzo Márquez (pasantes de Servicio Social de Cirugía Maxilofacial, Hospital General de Zacatecas "Luz González Cosío").
REFERENCES
AFFILIATIONS
1 Residente del Programa de Especialidad en Odontopediatría. Unidad Académica de Odontología. Universidad Autónoma de Zacatecas. Zacatecas, México.
2 Profesor en la Especialidad en Odontopediatría. Unidad Académica de Odontología. Universidad Autónoma de Zacatecas. Zacatecas, México.
3 Servicio de Cirugía Maxilofacial, Hospital General de Zacatecas "Luz González Cosío". Profesor de la Especialidad en Odontopediatría. Unidad Académica de Odontología. Universidad Autónoma de Zacatecas. Zacatecas, México.
CORRESPONDENCE
Cristal Yurixie Díaz Rosas. E-mail: yurixie_d@hotmail.comReceived: Marzo 2020. Accepted: Junio 2020.