Abstract
Central giant cell granulomas (CGCGs) of the jaw are benign intraosseous and osteolytic lesions. Their clinical presentation includes atypical aggressive lesions that cause severe damage early in life, and aggressive treatment is necessary in most cases to prevent recurrence. Curettage is the most preferred therapy, but in recent years, conservative therapy is also commonly used. Corticosteroid injection treatment, a conservative method, was planned for a developing male patient with difficulty in diagnosis and treatment due to the complications of resection. After clinical and radiologic evaluation of the patient, incisional biopsy was performed and the diagnosis of SDHG was made. The patient was treated with intralesional corticosteroids once a week for eight weeks. Since the patient’s treatment was interrupted for 2 months due to the Covid-19 outbreak, 2 doses of intralesional corticosteroid (Sinakort-A 40 mg) were administered and the patient was followed up. At 12 months, clinical and panoramic radiographic examination revealed no pathologic findings related to the lesion. In this article, we report a pediatric case of giant CGCG successfully treated with intralesional corticosteroid injection.
1
Introduction
Central giant cell granuloma (CGCG), which was first described by Jaffe [ ] in 1953, is a benign intraosseous lesion. It has a fibrotic lesion with multinucleated giant cells and a cluster of hemorrhagic areas [ ]. Jaffe [ ], for the first time, described CGCG as a traumatic bone cyst and fibrous dysplasia of the jaw bones secondary to bleeding or inflammatory processes; however, the term reparative was later abandoned [ , ]. Despite the current definition by the World Health Organization (WHO), the exact nature of the lesion is unclear [ ].
It accounts for nearly 7 % of all intraosseous lesions in the maxillofacial region [ ]. It most commonly affects the anterior mandibula in 60 % of individuals under 30 years of age [ ]. It is usually asymptomatic with a slow-growing pattern. The majority of patients present with painless swelling and fascial asymmetry [ ].
Imaging features of CGCG is usually non-specific and varies from ill-defined lesions to well-defined appearance. It typically appears as a unilocular or multilocular radiolucent lesion with smooth margins in the jaw bones. This type of granuloma can involve the mandible and maxilla, leading to migration of teeth and resorption of tooth roots, as well as expansion and destruction in the cortical bone. 9 It can be confused with other types of jaw bone lesions, as the radiographic appearance of CGCG is not pathognomonic. Both peripheral and central granulomas share similar histopathological features; both have cellular fibroblastic stromal cells, fibroblasts in the loose connective tissue, and few multinucleated giant cells disseminated in the capillary and connective tissue [ ]. In the differential diagnosis, ameloblastoma, primary and secondary hyperparathyroidism, fibrous dysplasia, cherubism, and aneurysmal bone cysts should be considered [ ]. In addition, CGCG cannot be always distinguished from the brown tumor of hyperparathyroidism histopathologically; therefore, further biochemical and endocrinological examinations may be required to establish a definitive diagnosis [ , ].
Treatment varies from local curettage to radical resection [ , ]. The recurrence rate increases up to 72 % following surgical curettage [ , ]. However, surgical resection is associated with low recurrence [ ]. Although radical surgery is an effective treatment modality, it may result in tooth loss, impaired inferior alveolar nerve function, and major defects which are difficult to reconstruct [ ]. Due to the lack of potential for malignant transformation, radiation treatment is contraindicated [ , ]. In addition, treatment with systemic calcitonin, intralesional corticosteroid injection or combined can yield successful outcomes [ ].
In this article, we report a pediatric case of CGCG who was successfully treated with intralesional corticosteroid injection.
2
Case report
A 10-year-old male patient was admitted to our clinic with swelling, pain, and asymmetry in the right side of the mandible. His medical history was non-specific without any systemic disease or trauma to the affected site. Extra-oral examination revealed no skin perforation or pus discharge. Intra-oral examination showed that expansion extended in the area between teeth 41 and 46. On palpation, bone stiffness was felt at the periphery of the mass and the stiffness decreased toward the center. Also, the swelling was in a hard consistency during palpation with pain in the relevant area, and the mucosa over the mass was hyperemic. Clinical examination revealed no nerve paresthesia related to the lesion in the chin. The preliminary diagnosis included a simple bone cyst, aneurysmal bone cyst, CGCG, or intraosseous hemangioma. Laboratory tests revealed normal values of calcium, phosphate, alkaline phosphatase, and parathyroid hormones.
Panoramic examination demonstrated a well-defined, unilocular radiolucent area extending between the mesial region of teeth 41 and 46 ( Fig. 1 ). The lesion caused resorption at the roots of teeth 83 and 84, pushing teeth 44 and 45 distally and tooth 43 mesially. Cone beam computed tomography (CBCT) was obtained from the relevant region to identify the true diameter and margin of the lesion. Axial, coronal, and sagittal CBCT images revealed a uniform radiolucent lesion with clear margins, starting from the mesial of tooth 41 and extending toward the mesial of tooth 45. The lesion expanded in the buccal and lingual cortical bone, perforated in the middle region of the lesion in the buccal and lingual cortical bone and depleted teeth 43, 44, and 45. Thinning and resorption areas were also seen in the buccal and lingual region of the bone ( Fig. 2 ). The initial intraoral photograph of the patient in Fig. 3 . The CBCT examination revealed a lesion size of 4.5 in the axial plane (mesiodistal), 3.2 cm in the axial plane (buccal-lingual) and 3.6 cm in the sagittal plane (cauda-spinal). For CBCT measurements, KaVo 3d eXam (KaVo Dental GmbH, Biberach, Germany) device was used, which is maintained and repaired annually at our institution. All images were obtained using the following technical specifications: 120 kVp, 5 mAs, 7 sec scan time, 0.125 mm voxel size, and 16 × 4 cm field of view (FOV).
Incisional biopsy was performed to the lesion. Histopathological examination revealed stromal cells with oval nuclei, spontaneous bleeding areas, and osteoclast type multinucleated giant cells. There were also typical mitoses in the stroma with fibrosis and ossification. Based on these findings, the patient was diagnosed with a CGCG consisting of spindle-shaped stromal cells.
No root canal treatment or surgical intervention was performed. Corticosteroid treatment containing triamcinolone acetonate (Sinakort-A 40 mg suspension for injection; İbrahim Etem Ulagay Menarini Pharmaceuticals, Istanbul, Turkey) was planned. Initially, eight doses of corticosteroid were administered intraorally once a week for a total of eight weeks. The treatment was applied at three different regions: i) distal to the lesion; posterior to anterior toward the center of the lesion; ii) mesial to the lesion; anterior to the posterior toward the center of the lesion; and iii) to the very center of the lesion. Once the first four doses of suspension for injection were administered, the bone islets formed inside the lesion were observed on the panoramic radiographs. Intraorally, areas of calcification in the form of bubbles were seen in the lesion area ( Fig. 4 ). Once eight doses of corticosteroid were administered, an increase in the bone islets was observed inside the lesion. Fig. 5 shows the patient’s panoramic radiograph during corticosteroid treatment. Since the patient was unable to be followed for two months due to the novel coronavirus disease 2019 (COVID-19) pandemic, two doses of the treatment were applied to the lesion at three points and the patient was, then, followed. At 12 months, clinical and panoramic radiographic examination revealed no pathological finding related to the lesion. At two years of follow-up, clinical and imaging studies showed a radiolucent area in the distal of the affected tooth 43 and surgical extraction in the horizontal position and the radiolucent lesion around it was indicated ( Fig. 6 ).
