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CASE REPORT
Ahead of print publication  

Central giant cell granuloma in a child: Surgical intervention by maintaining tooth germ


 Department of Pedodontics and Preventive Dentistry, Post Graduate Institute of Dental Sciences, Rohtak, Haryana, India

Date of Submission06-Feb-2020
Date of Decision10-Jun-2020
Date of Acceptance30-Jun-2020
Date of Web Publication04-Nov-2020

Correspondence Address:
Nisha Kumari,
782/20, Prem Nagar, Rohtak, Haryana
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/sjos.SJOralSci_8_20

  Abstract 


An intraosseous lesion of jaws called central giant cell granuloma (CGCG), is significantly found in children and young adults and most of the cases are nonaggressive, asymptomatic, and slow growing. Approximately 30% of cases show an aggressive and progressively devastating nature with tendency to recur. We present a case of slow growing CGCG in mandible in a 10-year-old male managed by curettage along with preservation of tooth germ.

Keywords: Central giant cell granuloma, curettage, nonaggressive



How to cite this URL:
Kumari N, Sarangal H, Beniwal D, Namdev R. Central giant cell granuloma in a child: Surgical intervention by maintaining tooth germ. Saudi J Oral Sci [Epub ahead of print] [cited 2020 Nov 25]. Available from: https://www.saudijos.org/preprintarticle.asp?id=299967




  Introduction Top


Central giant cell reparative granuloma was considered to be reactive-reparative in nature that might heal spontaneously and was first distinguished from other giant cell lesions of bones by Jaffe (1953).[1] According to the World Health Organization, central giant cell granuloma (CGCG) is defined as an intraosseous lesion comprising cellular fibrous tissue that include multiple foci of hemorrhage, collections of multinucleated giant cells and sometimes trabeculae of woven bone.[2] CGCG is a benign and rare lesion which usually occurs in children or young adults and mostly affects in the first 3 decades of life. It has a female predominance with a female to male ratio of 2:1.[3] A common location for CGCG occurrence is anterior part of mandible and has potential to cross midline.[2]

CGCG was classified as aggressive or nonaggressive by Choung et al. depending on the six criteria including presence of pain, enlargement of the lesion, expedition of growth, cortical bone perforation, root resorption of related teeth, and recurrence after removal. Nonaggressive lesions are slow growing, asymptomatic, less likely to dislocate teeth and usually discovered on routine X-ray, while aggressive lesions show rapid rate of growth and large in size usually >5 cm resulting in disturbance of facial symmetry, pain and numbness, tooth displacement, resorption of root, thinning, or perforation of cortical bone. A high recurrence rate has been reported after conservative surgical removal and resection may be needed later.[4]

CGCG may appear as unilocular or multilocular radiolucent defects, with welldefined or ill-defined margins radiographically with varying degrees of cortical expansion. Moreover, teeth displacement may be found in some cases. As radiographic findings are not evidential so may be confused with other lesions of the jaws.[3]

Treatment varies from simple curettage associated with conservative therapies to en bloc resection. However, recurrences rates are higher than 70% after surgical intervention.[5]


  Case Report Top


A 10-year-old boy presented to the department with painless swelling in the left mandibular canine-premolar region since 3 months which was not associated with any systemic symptoms. Medical and familial history was not significant. Extraoral examination was normal.

On intraoral examination, a diffuse swelling was present on the left side of mandible extending from first premolar to central incisor obliterating buccal vestibule. The swelling had smooth surface, was nontender and firm on palpation. The mandibular lateral incisor and canine were missing on the left side [Figure 1]a.
Figure 1: (a) Intraoral view showing swelling in relation to 33 and 34 tooth region, (b) intraoperative view, (c) after suturing, and (d) showing postoperative clinical view

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Occlusal radiograph revealed expansion of buccal cortical plate on the left side of mandible in canine premolar region [Figure 2]a and panoramic radiograph showed irregular radiolucent lesion with well-defined limits on the left side of mandible, extending from apical region of central incisor and first premolar. Mandibular left canine was displaced laterally and apically toward central incisor [Figure 3]a.
Figure 2: (a) Occlusal radiograph showing expansion of buccal cortical plate in relation to 31, 32, 33, and 34 tooth region and (b) showing postoperative follow-up view

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Figure 3: (a) Panoramic radiograph showing a well-defined radiolucency extending from left mandibular 1st premolar to central incisor and displaced canine. (b) Showing complete healing in the area of lesion and erupting canine

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Intraoral aspiration was attempted with 20 gauge needle under local anesthesia but did not yield aspirate, so radicular cyst, dentigerous cyst, and central hemangioma were ruled out. The patient was advised for investigations including serum calcium, phosphorus, alkaline phosphate, and parathhormone to exclude brown tumor of hyperparathyroidism. Fibrous dysplasia, aneurysmal bone cyst, and adenomatoid odontogenic tumor were also excluded because of peculiar radiographic findings. Hence, provisional diagnosis of unicystic ameloblastoma and CGCG was made.

Considering the clinical and radiographic findings, a surgical intervention was planned. The surgical site was infiltrated with 2% lignocaine with epinephrine. An incision was performed and a trapezoidal flap was raised to allow the complete excision of the lesion [Figure 1]b. The remaining bony cavity was thoroughly curetted, while maintaining the tooth germ, the bone margins regularized and the surgical area irrigated with buffered saline solution. The flap was reapproximated and closed with 3-0 silk sutures [Figure 1]c. Postsurgical instructions and antibiotic therapy were given to the patient.

After complete curettage, excised tissue was sent for histopathological examination which showed the presence of multinuclear giant cells, surrounded by mesenchymal ovoid and fusiform cells, with locations of blood extravasation, associated with pigmentation caused by the phagocytic hemosiderin [Figure 4], confirming the provisional diagnosis of CGCG.
Figure 4: High power photomicrograph (×40) showing stromal and multinucleated giant cells

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Sutures were removed after 7 days and a postoperative clinical picture, occlusal radiograph, and orthopantomography were performed [Figure 1]d, [Figure 2]b and [Figure 3]b Follow-up was done at 4 months' and 8 months' interval and osteoprotegerin (OPG) revealed complete healing of the lesion with no residual swelling. After excision of the lesion, the mandibular left canine got normalized position and began erupting at normal pace.


  Discussion Top


Various theories explaining the etiology and pathogenesis of CGCG have been proposed but its true nature is still unrevealed. It can be inflammatory or reactive in nature, a true tumor or an endocrine lesion. According to one hypothesis suggests that CGCG belongs to a range of mesenchymal proliferative vascular primary jaw lesions while in contrary to this, Vered et al. suggested small angiogenic activity stated that the low mean microvascular volume of vascular endothelial growth factor and basic fibroblast growth factor positive blood vessels.[6]

Histopathological examination of CGCG shows multinucleated giant cells along with mononuclear proliferating fibroblasts of polygonal or spindle shaped which are arranged in sheets and bundles in a collagenous stroma containing myxoid ground substance. Giant cells show pale vacuolated cytoplasm with variable number of nuclei (10–40). A variable amount of osteoid or newly formed bone beset CGCG along with extravasated red blood cells, hemosiderin-laden macrophages, and endothelial cell-lined vascular structures convoying proliferating fibroblasts that show congruence to granulation tissue.[4]

Treatment varies from conservative therapies like simple curettage to en bloc resection according to clinical signs and symptoms. Other treatment options for CGCG are intralesional administration of corticosteroids, systemic calcitonin, and antiangiogenic drugs. Nogueira et al. in 2012 conducted a study using intralesional triamcinolone, and reported good response in 71.43% (15 of 21 patients), however, 19.05% showed an average response (4 out of 21 patients), and a negative response in 9.52% (2 of 21 patients) was seen. Corticosteroids are cost-effective, ease to administer, and conservation of adjacent tissues, especially vital structures is the biggest advantage. However, they have to be used cautiously in patients suffering from diabetes, peptic ulcers, infections, and the immunocompromised or pregnant women.[7]

Calcitonin helps in bone deposition and hinders osteoclast activity, thereby suppressing bone resorption, so it can be used as an alternative to surgical treatment for aggressive CGCG which was first proposed by Professor Harris in 1993. Mode of the administration of calcitonin is subcutaneous route or nasal spray and the drawbacks comprise daily administration with long-term therapy, expensive. Moreover, it produces after effects such as nausea, vomiting, lightheadedness, and flushes.[4]

Systemic interferon alpha is comparatively a new therapeutic modality for CGCG. The conservative treatment along with interferon for prolonged period of time has shown good results and lead to conservation of teeth and vital structures. Interferon therapy is not used as parentage therapy as it remains a ferocious procedure and the adverse effects which must be considered are, bone marrow suppression and hair loss.[8]

The Food and Drug Administration in June 2013, accepted denosumab for treating CGCG as it inhibits osteoclastic activity by inhibiting receptor activation of nuclear factor kappa-B (RANK) ligand/OPG interaction and RANK. Schreuder et al. treated a young patient of CGCG with denosumab subcutaneous injections for 12 months after limitations of calcitonin and interferon treatment and concluded that aggressive CGCG lesions that did not answer to other remedies were effectively treated by denosumab, however to become a foundation therapy more studies are needed. Medication-related jaw osteonecrosis and osteomyelitis are the most severe side effect of denosumab.[9]

The maintenance of tooth germ is utmost important in children so, surgery is considered as the most accepted and conventional treatment. Eisenbud et al. proposed curettage or curettage plus peripheral osteotomy for the treatment of CGCG in children with the maintenance of tooth germs. Bataineh et al., treated 18 patients of aggressive CGCG with en bloc resection leaving 5 mm health tissue margin and reported recurrence in only one patient. According to most authors, conservative surgery is the only applicable procedure in children and young adults, as it facilitates spontaneous eruption of the tooth germs.[5]

Recurrence rate varies from 11% to 49% have been reported which may be the result of incomplete removal of crumbly, bleeding lesion, and incomplete excision in a larger-sized lesion which is more difficult to remove between teeth. According to Mooney et al., aggressive lesions has higher recurrence rate (72%) especially in young male patients.[9] Among recurring and nonrecurring CGCG, histological differences in the osteoid and distribution of giant cells was found statistically significant by Whitaker and Waldron and they concluded that uniform distribution of giant cells and lack of osteoid at their periphery is typically associated with recurrent lesions.[6]

In the present case, we planned for surgical treatment and curettage was performed under local anesthesia while preserving the tooth germ. The patient showed no signs of recurrence after 8 months of follow-up. After treatment, new bone deposition and eruption of displaced mandibular left canine started.

Therefore, conservative surgical treatment is a promising approach in children as it conserves the adjacent anatomical structures and avoid large surgical defect. Radiographic observation of CGCG can be performed on follow-up, considering the lesion has slow growth and less aggressive in nature. Hence, it should be diagnosed and managed at the earliest for minimizing the possible cost of esthetics, functional, and psychological problems.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Krishnappa S, Srinath S, Gopinath P, Krishnappa VS. Central giant cell granuloma in a 4-year-old female child. J Indian Soc Pedod Prev Dent 2015;33:337-40.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Patloth T, Reddy JS. Surgically treated central giant cell granuloma in six-year-old child: A case report. Int J Clin Pediatr Dent 2013;6:146-9.  Back to cited text no. 2
    
3.
Yuzbasioglu E, Alkan A, Ozer M, Bayram M. Multidisciplinary approach for the rehabilitation of central giant cell granuloma: A clinical report. Niger J Clin Pract 2014;17:528-33.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Elhag HA, Babikir MH, Tarakji B. Advantages and disadvantages of surgical and non-surgical treatment of central giant-cell granuloma: A review of literature. Int J Contemp Dent Med Rev 2017;2017:2.  Back to cited text no. 4
    
5.
Cavalcante RC, de Lucas Corso PF, Dias TR, Schramm E, de Souza PH, Rebellato NL, et al. Central giant cell granuloma (CGCG) in childhood: Surgical treatment by maintaining the tooth germs. RSBO Rev Sul Bras Odontol 2017;14:37-43.  Back to cited text no. 5
    
6.
Tenore G, Palaia G, Capocci M, Impellizzeri A, Romeo U. The central giant cells granuloma of the mandible: A report of two cases and a review of the literature. Senses Sci 2014;33.  Back to cited text no. 6
    
7.
Nogueira RL, Faria MH, Osterne RL, Cavalcante RB, Ribeiro RA, Rabenhorst SH. Glucocorticoid and calcitonin receptor expression in central giant cell lesions: Implications for therapy. Int J Oral Maxillofac Surg 2012;41:994-1000.  Back to cited text no. 7
    
8.
Goldman K, Marshall M, Alessandrini E, Bemstein M. Complications of alpha-interferon therapy for aggressive giant cell lesions of the maxilla. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:285-95.  Back to cited text no. 8
    
9.
Thomas D, Carriere P, Jacobs I. Safety of denosumab in giant-cell tumour of bone. Lancet Oncol 2010;11:815.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

 
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