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Authors: Dr. Balram Garg,Dr. Mohit Agarwal,Dr. Jatika Agarwal.

Central giant cell granuloma is an uncommon, benign intraosseous proliferative lesion with a female predilection, occurring under 30 years of age. Clinical features vary from a slow growing painless asymptomatic swelling to an aggressive lesion with pain, cortical perforation and root resorbtion. The management of CGCG will depend on the clinical and radiographic findings. The surgical treatment options includes local curettage, curettage with peripheral osteotomy or en-bloc resection .The medical management of CGCG as an adjunct to surgery includes intravenous administration with steroids or calcitonin, interferon-alpha, bisphosphonates, cryotherapy with liquid nitrogen and use of carnoy’s  solution.

Introduction –
Giant cell lesions are rare entity in maxillofacial region. Various giant cell lesions reported in literature specific to maxillofacial region are giant cell fibroma, cherubism, peripheral giant cell granuloma, central giant cell granuloma, giant cell tumor of bone, aneurysmal bone cyst, giant cell hyaline angiopathy, primary hyperparathyroidism, traumatic bone cyst, benign osteoblastoma and giant cell arteritis (tumoral arteritis). The histological, radiographic and clinical diagnosis is particularly difficult in these types of lesions due to their variable clinical behaviour and identical histological presentation with abundant giant cells. Among these, Central giant cell granuloma, also known as reparative giant cell granuloma, is an uncommon, benign intraosseous proliferative lesion with female predilection, occurring under 30 years of age. It was first introduced by Jaffe in 1953 to distinguish it from giant cell tumour of long bone.1 It is also known as Giant cell tumor of bone by some authors, while, others consider Giant cell tumor and Central giant cell tumor as different lesions2. We report here a case of Central giant cell granuloma of maxilla in a 29 year old female patient with clinical and radiographic features which was resembling a cyst.

Case Report –
A 29 years old lady reported to our centre with complain of change in contour of upper lip, elevation of left ala of nose and painless swelling of left midface region (fig 1) since four months. History reveals that swelling was started intra-orally and had gradually increased from small size to the present size. No history of trauma was elicited nor any systemic or local infections. Medical and family history was not significant.

 

   
FIGURE 1 PRE OPERATIVE FRONTAL PROFILE FIGURE 2 INTRA ORAL VIEW


Facial examination revealed a single diffused swelling over left midface region which was nontender and hard on palpation along with obliteration of left nasolabial fold. Overlying skin was appeared normal. Intraorally it was extending from midline in relation to upper left central incisor to left upper first premolar along with obliteration of left upper labial and buccal vestibular sulcus (fig 2). Overlying mucosa was appeared normal and all the involved teeth were vital. Swelling had smooth surface with no fluctuation, firm to hard in consistency and nontender on palpation. No tooth mobility and displacement was evident. Radiographically a single, multilocular radiolucent lesion with smooth margins was noticed extending from root tip of upper left central incisor to upper left first premolar region (fig 3).

 

   
FIGURE 3 OCCLUSAL RADIOGRAPH

 

FIGURE 4 INTRA-OPERATIVE VIEW

 

Roots tipping of lateral incisor and canine were also evident on radiograph. Aspirates from the lesion yields blood tinged fluid, so based on clinical and radiological finding lesion was provisionally diagnosed a cystic lesion with the differential diagnosis of Ameloblastoma, Odontogenic keratocyst, Globulomaxillary cyst, adenomatoid odontogenic tumor and Giant cell lesion. Surgical excision of lesion under local anaesthesia was planned and the mass was excised (figure4) without complications.

   
FIGURE 5 HISTOPATHOLOGICAL PHOTOMICROGRAPH

 

FIGURE 6 POST OPERATIVE FRONTAL VIEW

 

Excised mass was sent for histopathological examination. Histopathological examination confirmed the diagnosis of giant cell lesion as Central gaint cell granuloma. The histopathological examination showed a highly vascularized fibrous stroma with several multinucleated giant cells with 20-30 nuclei. Randomly dispersed spindle shaped fibroblasts were also noted (fig 5). Patient has been followed up for 01 year post-operatively with no sign of recurrence. Patient upper lip and ala contour have been restored to normal (fig 6).

Discussion- Central giant cell granuloma is a nonneoplastic proliferative lesion of unknown etiology. Various theories brand it from being a 'reactive' to hamartomatous to a neoplastic lesion (Pogrel 2004)3.It has now been hypothesized that it is the mononuclear spindle shaped cell which controls the proliferative activity of this lesion, as opposed to the more frequently seen giant cell.4 They are defined by the World Health Organization as an intraosseous lesion consisting of cellular fibrous tissue containing multiple foci of haemorrhage, aggregations of multinucleated giant cells, and occasionally, trabeculae of woven bone.5 The central giant cell granuloma (CGCG) of the jaws accounts for approximately 7% of all benign tumors of the jaws (Kramer et al, 1991). Lesions occur more frequently in the mandible than in the maxilla in the anterior region of the jaws (Regezzi & Scuibba, 1989). The present case, however, involved the maxilla. Lesions are found predominantly in children and young adults, with most cases (as high as 75%) presenting before 30 years of age. Females are affected more often than males, in a ratio of 2 to 1. Clinically it may be manifested from as a slow growing painless asymptomatic swelling to a aggressive lesion with pain, cortical perforation and root resorbtion6 The most common presenting sign of CGCG is a painless swelling with noticeable facial asymmetry.5 This case article also presents the case of female patient aged 29 years old with painless asymptomatic swelling. The radiological appearance of CGCG is variable. The CGCG may occur initially as a unilocular, cyst-like radiolucency, but as it grows larger, it frequently develops an architecture that causes a soap-bubble type of multilocular radiolucency. It may be well-defined or ill-defined and shows variable expansion and destruction of the cortical plate.7 This radiographic feature could also be well correlated with the present case as a single, multilocular radiolucent lesion with smooth margins. The radiological appearance of the lesion is not pathognomonic and may be confused with that of many other lesions of jaws.8 The final diagnosis eventually rests on histopathology because the clinical and radiological features are not specific. In the present case report also provisional diagnosis of a cystic lesion was made based on clinical and radiological finding and final diagnosis of Central giant cell granuloma was confirmed only after histopathological examination. Other differential diagnosis included Ameloblastoma, Odontogenic keratocyst, Globulomaxillary cyst, adenomatoid odontogenic tumor and Giant cell lesion which were excluded after histopathological examination. Histologically, CGCG contain focal arrangements of giant cells within a vascular stroma with thin-walled capillaries adjacent to the giant cells10. There is a spindle cell stroma which may well be the cell of origin10. These findings were consistent with diagnosis of CGCG. It is difficult to distinguish this lesion histologically from brown tumor of hyperparathyroidism. Hence serum levels of calcium, phosphorus and alkaline phosphatase were advised which were found to be within the normal limits thus establishing the diagnosis of central giant cell granuloma9. The management of CGCG will depend on the clinical and radiographic findings.11 The traditional treatment of CGCG has been local curettage. However, aggressive sub-types of CGCG show a tendency to recur and necessitate bone resection that may determine extensive defects in jaws. The medical management of CGCG as an adjunct to surgery includes treatment with steroids or calcitonin, interferon-alpha, bisphosphonates have been administered intravenously, cryotherapy with liquid nitrogen and use of carnoy solution with promising results.3 In the present case lesion was surgically excised and satisfactory results with no recurrence were found on regular follow up of the patient.

Conclusion
The clinical behaviour of this lesion is quite variable and difficult to predict. These lesions are not reported so frequently in literature and thus can be misdiagnosed as cyst or tumor. In this case report, also, lesion presented as odontogenic cyst or tumor. The lesion was confirmed as Central giant cell granuloma only after confirmation from histopathological examination. Clinical and radiological features of odontogenic cysts and tumors should be studied carefully to differentiate them from Central giant cell granuloma.

References

  1. Jaffe HL. Giant cell reparative granuloma, traumatic bone cyst, and fibrous (fibroosseous) dysplasia of the jaw bones. Oral Surg 1953;6:159-75.
  2. Auclair PL, Cuenin P, Kratochvil FJ, et al: A clinical and histomorphologic comparison of the central gaint cell granuloma and the gaint cell tumor, Oral Surg Oral Med Oral Pathol 66:197-208, 1988.
  3. Regezi JA, Pogrel MA. Comments on the pathogenesis and medical treatment of central giant cell granulomas (letter). J Oral Maxillofac Surg 2004; 62: 116-8
  4. O'Malley M, Pogrel MA, Stewart JC et al. Central giant cell granulomas of the jaws: Phenotype and proliferation-associated markers. J Oral Pathol Med 1997; 26: 159.
  5. Barnes L, Eveson JW, Reichart P, Sidransky D, eds. Pathology and Genetics of Head and Neck Tumours. Kleihues P, Sobin LH, series eds. World Health Organization Classification of Tumours. Lyon, France: IARC Press, 2005. p. 324.
  6. Chuong R, Kaban LB, Kozakewich H, Perez-Atayde A. Central giant cell lesions of the jaws: a clinicopathologic study. J Oral Maxillofac Surg 1986;44:708-13.
  7. Kaffe I, Ardekin L, Taicher S, et al: Radiologic features of central gaint cell granuloma of the jaws, Oral Surg Oral Med Oral Pathol Oral Radio Endod 81:720-726, 1996
  8. Wood NK, Goaz PW, Kallal RH. Multilocular radiolucencies. In: Wood NK, Goaz PW, eds. Differential diagnosis of oral and maxillofacial lesions. 5th ed. St. Louis: Mosby. 1997; 333- 55.
  9. Neville BW, Damm DD, Alien CM, Bouquot JE, editors. Oral & Maxillofacial Pathology. 2'd ed. Philadelphia: Saunders 2002: 544-6.
  10. Liu B, Yu SF, Li TJ. Multinucleated giant cells in various forms of giant cell containing lesions of the jaws express features of osteoclasts. J Oral Pathol Med 2003; 32: 367.

More references are available on request

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