Users Online: 279
Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Login 

 Table of Contents  
LETTER TO THE EDITOR
Year : 2020  |  Volume : 9  |  Issue : 1  |  Page : 58-67

Isolated regional nodal metastasis in giant cell tumor of the bone: Case report and review of literature


Department of Medical Oncology, Tata Memorial Hospital, Mumbai, Maharashtra, India

Date of Web Publication26-Dec-2019

Correspondence Address:
Dr. Anand Raja
Department of Surgical Oncology, Chennai, Tamil Nadu, India
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sajc.sajc_244_19

Rights and Permissions

How to cite this article:
Malik K, Raja A, Shirley S. Isolated regional nodal metastasis in giant cell tumor of the bone: Case report and review of literature. South Asian J Cancer 2020;9:58-67

How to cite this URL:
Malik K, Raja A, Shirley S. Isolated regional nodal metastasis in giant cell tumor of the bone: Case report and review of literature. South Asian J Cancer [serial online] 2020 [cited 2020 Jul 12];9:58-67. Available from: http://journal.sajc.org/text.asp?2020/9/1/58/274057

Dear Editor,

Giant cell tumors (GCTs) of the bone are benign osteolytic tumor commonly occurring in the third to fourth decades with distal femur being the most common site.[1] They form 5% of the primary skeletal tumors.[2] Metastasis though uncommon is usually seen with recurrences. Curettage with intralesional adjuvant therapy with polymethylmethacrylate, liquid nitrogen, and bone graft has been successfully used to reduce recurrences while preserving function.[3] The management of metastasis is still debatable due to the scarcity of literature.

A 47-year-old female who was diagnosed to have GCT of the left radius for which she underwent left distal radius curettage with bone grafting. After 1 year of disease-free interval (DFI), she developed local recurrence and underwent en bloc resection of radius with reconstruction using nonvascularized fibula graft with plating. She developed local recurrence in the soft tissue in the next 2 years which was managed by wide local excisions. After a DFI of 1 year, she presented with an enlarged mobile epitrochlear node, fine-needle aspiration cytology of which was suggestive of GCT. Computed tomography of the chest showed no pulmonary metastasis. The patient was discussed in multispecialty board and she underwent epitrochlear nodal dissection. The final histopathology showed five nodes, with one node showing metastatic GCT. There was no perinodal spread. [Figure 1] and [Figure 2] show the presence of tumor within the lymph nodes. She is on regular follow-up for 1 year and has shown no sign of recurrence.
Figure 1: Lymph node replaced by metastatic tumor composed of aspatially arranged multinucleate giant cells in a background of mononuclear stromal cel l s (H and E, ×40)

Click here to view
Figure 2: Higher magnification shows uniformly arranged multinucleate giant cells admixed with mononuclear stromal cells with residual lymphoid follicle (right extreme) (H and E, ×100)

Click here to view


GCT is characterized by scattered multinucleate giant cells among mononuclear stromal cells, together imparting a syncytium-like appearance causing a typical radiolucent lytic shadow on X-ray.[4] The tumor is known for its propensity for local recurrences which are generally amenable to wide excisions. The potential for metastatic spread was first reported by Jaffe et al. in 1940.[5] Distant metastasis is seen in 2%–3% of cases with the lungs being the most common site of distant metastasis and the incidence of pulmonary metastasis varies from 4% to 11% in the literature.[6] Other uncommon sites being the bone, skin, soft tissue, breast, and endobronchial tree have also been reported.[7]

Lymph nodal involvement is very rare in GCT and only 13 cases have been reported in the English literature.[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19] [Table 1] lists the cases of GCT with lymph node involvement. Dyke, in 1931, reported the presence of GCT in lymph nodes in a case with extensive metastatic disease.[8] Since then, majority have reported mediastinal and para-aortic nodal involvement. Only five have reported regional lymph node involvement, of which two were associated with pulmonary metastasis.[7],[9],[10],[11],[12] Isolated regional node involvement as seen in our case has been reported previously only in three cases.[9],[10],[11] Budzilovich et al. reported the first regional nodal involvement in 1963.[9] Present et al., in 1986, reported a GCT metastasizing to regional lymph node; however, it spread to lungs after a year.[10] In another case reported by Aftab and Umar, there was axillary nodal spread from GCT of the distal humerus without pulmonary involvement.[11]
Table 1: Cases of giant cell tumor of bone with lymph node metastasis

Click here to view


In one of the largest reviews from a tertiary care center, Viswanathan and Jambhekar retrospectively evaluated 470 patients of GCTs, of which 24 had distant metastasis and only one patient had regional lymph node metastasis to inguinal lymph node. In their patient, the primary site was femur and there were multiple lung metastasis along with lymph node metastasis. The authors concluded that there was no association between clinicopathological variables and the development of metastasis.[12]

The mechanism of pulmonary metastasis has been speculated to be tumor emboli either upfront or during the time of curettage. Lymphatic spread can also be explained similarly by the multiple surgeries performed previously for our patient.

Metastatic GCT has been successfully treated with complete resection of primary and the metastasis. Pulmonary metastatectomy has been routinely performed in resectable patients, with similar principle applied for lymph node metastasis also. All four cases with regional lymph node metastasis have been managed with surgical resection. Connell et al. successfully managed a patient with excision of primary patellar GCT along with excision of posterior mediastinal nodal mass.[13] Another similar case of GCT of the patella with pulmonary and nodal metastasis treated with metastasectomy.[7] Alternate treatments for unresectable disease have also been attempted with limited success. Lewis et al., in 1996, reported a case of distal radius GCT with mediastinal node metastasis. They used high dose steroids to downsize the tumor followed by resection of the mediastinal mass.[14] There are isolated cases where chemotherapy and radiotherapy have been utilized with varied success rates.[18] In the absence of literature, no definitive recommendation can be made.

In the present case, an upfront incomplete surgery followed by multiple recurrences with repeated surgeries would have led to tumor emboli spreading through the lymphatics. The patient was successfully managed by lymph node dissection. The implication of lymph node and distant spread in malignant GCT is not as worse compared to other malignancies and hence should be treated with curative intent if completely resectable.

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.
Turcotte RE. Giant cell tumor of bone. Orthop Clin North Am 2006;37:35-51.  Back to cited text no. 1
    
2.
Neff JR. Primary tumours of bone. An overview in adults. In: Moosa AR, Schimpff SC, Robson S, editors. Comprehensive Textbook of Oncology. 2nd ed. Baltimore: Williams and Wilkins; 1991. p. 1168-70.  Back to cited text no. 2
    
3.
Kivioja AH, Blomqvist C, Hietaniemi K, Trovik C, Walloe A, Bauer HC, et al. Cement is recommended in intralesional surgery of giant cell tumors: A Scandinavian Sarcoma Group study of 294 patients followed for a median time of 5 years. Acta Orthop 2008;79:86-93.  Back to cited text no. 3
    
4.
Werner M. Giant cell tumour of bone: Morphological, biological and histogenetical aspects. Int Orthop 2006;30:484-9.  Back to cited text no. 4
    
5.
Jaffe HL, Lichtenstein L, Partis RB. Giant cell tumour of bone: Its pathological appearance, grading, supposed variants and treatment. Arch Path 1940;30:993-1031.  Back to cited text no. 5
    
6.
Muheremu A, Niu X. Pulmonary metastasis of giant cell tumor of bones. World J Surg Oncol 2014;12:261.  Back to cited text no. 6
    
7.
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. 7
    
8.
Dyke SC. Metastasis of the “benign” giant cell tumor of bone (osteoclastoma). J Pathol 1931;34:259.  Back to cited text no. 8
    
9.
Budzilovich GN, Truchly G, Wilens SL. Tumor giant cells in regional lymph nodes of a case of recurrent giant cell tumor of bone. Clin Orthop Relat Res 1963;30:182-7.  Back to cited text no. 9
    
10.
Present DA, Bertoni F, Springfield D, Braylan R, Enneking WF. Giant cell tumor of bone with pulmonary and lymph node metastases. A case report. Clin Orthop Relat Res 1986;209:286-91.  Back to cited text no. 10
    
11.
Aftab K, Umar B. Giant cell tumor of bone with lymph node metastases: A rare presentation. Indian J Pathol Microbiol 2011;54:396-7.  Back to cited text no. 11
[PUBMED]  [Full text]  
12.
Viswanathan S, Jambhekar NA. Metastatic giant cell tumor of bone: Are there associated factors and best treatment modalities? Clin Orthop Relat Res 2010;468:827-33.  Back to cited text no. 12
    
13.
Connell D, Munk PL, Lee MJ, O'Connell JX, Janzen D, Vu M, et al. Giant cell tumor of bone with selective metastases to mediastinal lymph nodes. Skeletal Radiol 1998;27:341-5.  Back to cited text no. 13
    
14.
Lewis JJ, Healey JH, Huvos AG, Burt M. Benign giant-cell tumor of bone with metastasis to mediastinal lymph nodes. A case report of resection facilitated with use of steroids. J Bone Joint Surg Am 1996;78:106-10.  Back to cited text no. 14
    
15.
Rock MG, Pritchard DJ, Unni KK. Metastases from histologically benign giant-cell tumor of bone. J Bone Joint Surg Am 1984;66:269-74.  Back to cited text no. 15
    
16.
Kay RM, Eckardt JJ, Seeger LL, Mirra JM, Hak DJ. Pulmonary metastasis of benign giant cell tumor of bone. Six histologically confirmed cases, including one of spontaneous regression. Clin Orthop Relat Res 1994; 302:219-30.  Back to cited text no. 16
    
17.
Goldenberg RR, Campbell CJ, Bonfiglio M. Giant-cell tumor of bone. An analysis of two hundred and eighteen cases. J Bone Joint Surg Am 1970;52:619-64.  Back to cited text no. 17
    
18.
Vanel D, Contesso G, Rebibo G, Zafrani B, Masselot J. Benign giant-cell tumours of bone with pulmonary metastases and favourable prognosis. Report on two cases and review of the literature. Skeletal Radiol 1983;10:221-6.  Back to cited text no. 18
    
19.
Sung HW, Kuo DP, Shu WP, Chai YB, Liu CC, Li SM. Giant-cell tumor of bone: Analysis of two hundred and eight cases in Chinese patients. J Bone Joint Surg Am 1982;64:755-61.  Back to cited text no. 19
    


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
References
Article Figures
Article Tables

 Article Access Statistics
    Viewed282    
    Printed1    
    Emailed0    
    PDF Downloaded84    
    Comments [Add]    

Recommend this journal