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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 2  |  Issue : 1  |  Page : 12-14

Osteoclastoma at the maxillofacial region


1 Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha ‘O’ Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India
2 Medical Research Laboratory, IMS and SUM Hospital, Siksha ‘O’ Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India

Date of Web Publication27-Nov-2018

Correspondence Address:
Dr. Santosh Kumar Swain
Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha ‘O’ Anusandhan (Deemed to be University), K8, Kalinga Nagar, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/aiao.aiao_16_18

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  Abstract 


Osteoclastoma or giant cell tumor is an uncommon neoplasm of the bone. Common sites for osteoclastomas are long bones. Osteoclastoma of the craniofacial bones is extremely rare clinical entity. Here, we are reporting a case of osteoclastoma originating from the left maxilla presenting with swelling at the left nasolabial area with nasal obstruction. He had undergone complete excision of the tumor under general anesthesia. During the postoperative period, the patient did well without any evidence of recurrence or metastasis for 1-year follow-up.

Keywords: Giant cell tumor, maxilla, maxillofacial region, osteoclastoma


How to cite this article:
Swain SK, Bhattacharyya B, Sahu MC. Osteoclastoma at the maxillofacial region. Ann Indian Acad Otorhinolaryngol Head Neck Surg 2018;2:12-4

How to cite this URL:
Swain SK, Bhattacharyya B, Sahu MC. Osteoclastoma at the maxillofacial region. Ann Indian Acad Otorhinolaryngol Head Neck Surg [serial online] 2018 [cited 2018 Dec 14];2:12-4. Available from: http://www.aiaohns.in/text.asp?2018/2/1/12/246142




  Introduction Top


Osteoclastoma or giant cell tumor is a benign neoplasm of the bone but often locally aggressive in nature. It is usually seen between the third and fifth decades of life. It is sometimes seen after 50 years of age, whereas it is rarely seen after 60 years of age. This is one of the bony tumors showing higher prevalence among females.[1] It accounts for about 20% of benign bone tumors and approximately 5% of the biopsied primary bone neoplasms.[1] Osteoclastoma is often seen near the articular end of the tubular bones. The bony part of the head-and-neck region like maxillary bone is rarely affected by osteoclastoma. It has tendency for recurrence and delayed malignant transformation with metastases, particularly to the lung, has been reported.[2] The preferred modality of treatment is radical surgical excision. Due to rarity of this lesion and presentation, we describe a case of osteoclastoma arising from the maxillofacial area involving maxillary bone and affecting the nasal cavity and producing the bulging at hard palate.


  Case Report Top


A 45-year-old female attended the outpatient department of otolaryngology for progressive swelling of the left maxillofacial area for 4 months with a complaint of left nasal obstruction for 3 months. On examination, there was swelling at the left maxillofacial area and left palatal region [Figure 1]. The mass was hard in consistency, nontender, and fixed. The skin over the mass was normal. The local temperature over the mass was normal. The rest of the head-and-neck regions were within normal limit. Computed tomography scan showed lobulated mass at the left alveolar ridge and hard palate with cortical expansile mass with little protrusion toward the nasal cavity and maxillary sinus [Figure 2]. Fine-needle aspiration cytology was done from nasofacial bulging which revealed osteoclast-like giant cells with oval nucleus. Serum calcium was normal whereas serum alkaline phosphatase was increased (290 μ/L). Surgery was planned for excision of the mass under general anesthesia. The mass was excised through the sublabial approach where the mass was extending from the hard palate to lateral wall of the nose and upper left alveolar ridge. The excised mass was sent for histopathological examination. The gross appearance of the excised mass was irregular mass of grayish appearance of 2.5 cm × 1.5 cm × 5 cm. The microscopic picture of the mass revealed large multinucleated giant cells, osteoclasts, stromal cells, and inflammatory cells [Figure 3]. The histopathological examination of the excised mass confirmed the diagnosis of osteoclastoma or giant cell tumor. The postoperative period of the patient was uneventful with no evidence of recurrence during 1-year follow-up.
Figure 1: Clinical picture showing at left maxillofacial area

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Figure 2: CT scan picture showing mass with typical soap bubble appearance at left lower alveolar area

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Figure 3: Microphotograph showing multinucleated osteoclast types giant cells and multiple monocytes arranged in compact fashion. (Hematoxylin and eosin stain)

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  Discussion Top


Osteoclastoma or giant cell tumor is an uncommon primary bony tumor which often seen in the long bones. It is a benign neoplasm but sometimes locally aggressive in nature. There is the presence of multinucleated giant cells (osteoclast-like cells) in this tumor. The cytological description of osteoclastoma was first documented by Cooper and Traverse in 1818, and its potential for malignant transformation was described by Virchow.[3] The common sites for this tumors are epiphysis of long bones such asdistal femur, distal part of radius, and proximal part of the tibia.[4] Osteoclastoma or giant cell tumors constitute 4%–5% of all primary bony neoplasms.[5] More than 75% of the osteoclastomas are seen in the epiphyseal part of the long bones and common sites in the order of frequency are lower part of the femur, upper part of the tibia, and lower part of the radius. Around 2% of all the osteoclastoma arise in the head-and-neck area, with majority of them seen in the sphenoid sinus, ethmoidal sinus, and temporal bones.[4] Osteoclastoma contains multinucleated giant cells (osteoclast-like cells) which is characteristic of this neoplasm. Malignancy in osteoclastoma is rare and seen in 2% of cases. The giant cell tumor stromal cells are neoplastic cells which arise from the osteoblastic origin and express different osteoblast cell markers such as alkaline phosphatase and osteocalcin.[6] The mononuclear phagocyte cells present in different parts of the body such as in the vascular system are called monocytes and that are present in other parts are called macrophages. The potent chemoattractic factor for monocytes is called as monocytic chemoattractant protein 1 (MCP-1). The isolation and regulation of MCP-1 mRNA and protein in osteoclastoma can be done using Northern blot analysis, in situ hybridization, and immunohistochemistry. Immunohistochemistry and in situ hybridization show MCP-1 gene transcription and protein which consistently seen in cytoplasm of stromal-like tumor cells of osteoclastoma.[6] It is usually seen in 30–50 years of age group where 16% of the patients are under the age of 20 years.[3] Patients of osteoclastoma often present with slowly progressive swelling with or without pain. Clinical symptoms arise when the tumor begins to damage the cortex and irritate the periosteum. When tumor causes weakening of the bone, it leads to pain and pathological fracture. The tumor appears as red and areas of collagens seen as gray color. It is more commonly seen in bones which develop by endochondral ossification and rare in the head-and-neck region. These tumors are often difficult to differentiate from benign lesions of the nasofacial area and other malignant lesions. The differential diagnoses of osteoclastoma are giant cell reparative granuloma, osteoblastoma, Brown tumor of hyperparathyroidism, aneurysmal bone cyst, osteoblastoma, nonossifying fibroma, benign fibrous histiocytoma, and foreign body reaction.[7] Osteoclastoma is a benign tumor but can be locally aggressive. It rarely metastasizes to other parts of the body. The diagnosis of this tumor is based on the biopsy and imaging. On gross examination, the tumor appears osteolytic expansion with bony destruction and thinning of the cortex. The tumor shows focal areas of necrosis, cystic formation, and hemorrhage. Histopathological picture shows multinucleated giant cells, osteoclasts with up to hundred nuclei which have prominent nucleoli. Multinucleated osteoclast-type giant cells and mononuclear cells in the mesenchymal tumor are arranged in compact fashion. The mononuclear cells which present in the tumor show round to oval nuclei with uniformly presence of chromatin and indistinctly seen nucleoli with eosinophilic cytoplasm. The multinucleated giant cells in the tumor mass are uniformly distributed and possess a variable number of nuclei, often 40–60 nuclei. The mononuclear cells are spindle shaped and arranged in storiform fashion. However, there is a poor correlation between histological picture and chance to recur or malignant transformation. Complete surgical excision with long-term follow-up is the treatment of choice for managing the osteoclastoma of the maxillofacial region. Curettage is often used technique for this lesion. Curettage and acrylic cementation are helpful in case of large giant cell tumor. Complete excision of the tumor was done in our case. The treatment of osteoclastoma of the head-and-neck region is controversial. It is often managed surgically although it has nature to recur. Few authors recommend postoperative radiotherapy. The indications of radiotherapy in osteoclastoma are inoperable and incomplete rejected tumor and in case of recurrence after surgery.[8] Many drugs are also prescribed in osteoclastoma and these are bisphosphonates such as Zoledronate, which may induce apoptosis multinucleated giant cell fraction and prevent tumor-induced osteolysis. Zoledronate is effective for killing osteoclast-like cells as in vitro studies.[9] Recently, humanized monoclonal antibodies such as denosumab targeting the RANK ligand are used for the treatment of osteoclastoma. There is thought that increased expression of RANK ligand by stromal cells which play important role in tumorigenesis.[10] The recurrence rate is quite high approximately 40%–60% seen within the first 2 years after treatment of the osteoclastoma.[11] Although osteoclastoma is rare, it should be considered as one of the differential diagnoses in case of any swelling at the maxillofacial area.


  Conclusion Top


Osteoclastoma at the maxillofacial area is unusual with respect to its location of origin. Although rare in the maxilla, it should be considered as one of the differential diagnoses of swelling at the maxillofacial area or maxillary swelling. The diagnosis of this tumor is done on the basis of imaging and histopathological examinations. Adequate surgical excision of the tumor and long-term follow-up are essential for managing the osteoclastoma at the nasofacial area.

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.
Quadros RS, de Sousa AA, Carvalho GT, Dellaretti Filho MA. Multifocal osteoclastoma of the skull: Case report. Arq Neuropsiquiatr 2004;62:167-9.  Back to cited text no. 1
    
2.
Qureshi SS, Puri A, Agarwal M, Desai S, Jambhekar N. Recurrent giant cell tumor of bone with simultaneous regional lymph node and pulmonary metastases. Skeletal Radiol 2005;34:225-8.  Back to cited text no. 2
    
3.
Venkatesh MD, Vijaya N, Girish N, Galagali JR. Giant cell tumor of temporal bone: A case report. Med J Armed Forces India 2012;68:392-4.  Back to cited text no. 3
    
4.
Singh HP, Kumar S, Aga P, Kumar M, Usmani SA, Agarwal SP. Huge osteoclastoma of hyoid bone: A case report. Indian J Otolaryngol Head Neck Surg 2016;68:123-5.  Back to cited text no. 4
    
5.
Som PM, Brandwein M. Sinonasal cavities: Inflammatory diseases, tumors, fractures and postoperative findings. In: Som PM, Curtin HD, editors. Head and Neck Imaging. 3rd ed., Vol. 1. New York: C.V. Mosby Year Book Publishers; 1996. p. 126-318.  Back to cited text no. 5
    
6.
Huang L, Xu J, Wood DJ, Zheng MH. Gene expression of osteoprotegerin ligand, osteoprotegerin, and receptor activator of NF-kappaB in giant cell tumor of bone: Possible involvement in tumor cell-induced osteoclast-like cell formation. Am J Pathol 2000;156:761-7.  Back to cited text no. 6
    
7.
Wieneke JA, Gannon FH, Heffner DK, Thompson LD. Giant cell tumor of the larynx: A clinicopathologic series of eight cases and a review of the literature. Mod Pathol 2001;14:1209-15.  Back to cited text no. 7
    
8.
Malwar MM. Link MP. Sarcoma of bone. In: Devita VT Jr., Hellman S, Rosenberg SA, editors. Cancer, Principles and Practice of Oncology. 5th ed. Philadelphia: Lippincott-Raven; 1992. p. 1816-52.  Back to cited text no. 8
    
9.
Thomas DM, Skubitz KM. Giant cell tumour of bone. Curr Opin Oncol 2009;21:338-44.  Back to cited text no. 9
    
10.
Agrawa A, Shukla A. “Osteoclastoma of maxilla” a rare case. Indian J Otolaryngol Head Neck Surg 2016;68:104-6.  Back to cited text no. 10
    
11.
Iizuka T, Furukawa M, Ishii H, Kasai M, Hayashi C, Arai H, et al. Giant cell tumor of the temporal bone with direct invasion into the middle ear and skull base: A case report. Case Rep Otolaryngol 2012;2012:690148.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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