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CASE REPORT

 Metastatic Navicular Giant Cell Tumour (Osteoclastoma)- A Case Report With 16 Year Follow-Up

Yasir Khan*, Moore D P *

* Our Lady’s Hospital for Sick Children, Crumlin,  Dublin 12,  Republic of Ireland.

Address for Correspondence:

Dr Yasir Khan
Orthopaedic registrar,
Our lady’s Hospital, Navan,
Republic of Ireland.
Mobile
: 00353-86-608 8273
E-mail: ymkhan70@gmail.com

 

Abstract:

Giant cell tumour or osteoclastoma is a locally osteolytic tumour which rarely spread. It most frequently occur in the epiphysis of the distal femur or proximal tibia, predominantly affecting skeletally mature young adults. 

      We present a 16 year follow-up of a case of navicular osteoclastoma who presented due to pulmonary lesions.     

      The primary site was in the navicular. The first presentation of the patient was at the age of 14, when he presented with shortness of breath in a peripheral hospital. He was then diagnosed and referred to our institution. He was treated initially with chemotherapy for pulmonary lesions; primary site was treated with en-bloc resection of navicular with lower pole of medial cuneiform. 

       There was recurrence of pulmonary lesions three years after initial presentation which was treated with, two stage thorocotomy. There was no local bony recurrence. 

       So far there is no agreement on how to treat patients of osteoclastoma who present with pulmonary metastases. Our protocol to treat this patient with chemotherapy for pulmonary lesion and excision of the lesion at primary site proved effective, though larger studies required to support this.   

J.Orthopaedics 2007;4(4)e21

Keywords:  Navicular, Giant cell tumour, Pulmonary metastasis, Osteoclastoma

.
Introduction:
 

       We report a 16 year follow-up of a case of navicular osteoclastoma, presented due to pulmonary lesion. We discuss the course of the disease, its symptoms and signs, treatment and final outcome.

Case Report :

13 year old boy presented in November 1987 in another institution with sudden onset of pleuritic central chest pain with dyspnoea on exertion and mild dyspnoea at rest. He had complained of painless swelling in his right ankle for the last 2 years. The swelling gradually increased in size. Two months prior to his presentation he sprained his right ankle and since then he had complained of some discomfort in the ankle. 

       He was found on chest X-ray to have a large right sided pleural effusion with bilateral multiple pulmonary lesions with more extensive areas of abnormal density, in the right lower lobe, posteriorly (figure 1 & 2). A foot X-ray showed a lytic Lesion, in the right navicular bone (figure 2 & 3). A biopsy of the navicular revealed well Differentiated, Giant cell tumour. He was transferred to our institution for further investigation, and management. 

           An open lung biopsy was performed, the histology confirmed the presence of Malignant tissue, identical to that of the lesion in the navicular bone. The diagnosis of Metastatic giant cell tumour (osteoclastoma) of the navicular bone was made.                                                                                   

           A literature search was un-helpful as there was no reported case at that time, of patient’s with osteoclastoma, presenting with pulmonary metastases. It was decided to treat him as malignant sarcoma and he was commenced on chemotherapy. The agents which were used include Ifofosfamide, Adriamycin, Vincristine, Carboplatinum, and VP16. After the initial course of chemotherapy he developed severe neutropenia but recovered. His pleural effusion improved and his pulmonary lesions gradually resolved but there was no change in the size of the primary lesion.  

           He received his last course of chemotherapy, in March 1988.Because of ongoing discomfort in his right foot, in November 1988, an en-bloc resection of right navicular bone with lower pole of medial cuneiform was performed.  

           He remained well for a period of 3 years, when he developed a recurrence of his pulmonary lesions, bilaterally, which was evidenced by increase in the size of the previous lesions. It was treated with two stage thorocotomy with resection of the pulmonary Lesions. He did not receive chemotherapy. 

           He was recently reviewed in outpatients 16 years after initial presentation. He is not reporting any problems, apart from some discomfort in the right foot after running for 3-4 miles. Recent X-rays of right foot showed no recurrence of primary lesion and chest radiograph showed residual calcified nodules (figure 4) for which he has no respiratory problems.

Figure 1: Right side pleural effusion with bilateral pulmonary metastasis

Figure 2: histology slide of pulmonary lesion

 

Figure 3: Same patient with right navicular giant cell tumour

Figure 4: HISTOLGY of Navicular lesion

Figure 5: Recent chest X-ray showing residual pulmonary nodules

Discussion:

          Giant cell tumour has been described as a benign tumour which gives rise to lytic lesions in the bones and has the rare ability to spread. It affects usually the epiphysis of the distal femur or proximal tibia, predominantly affecting skeletally mature young adults. Pulmonary lesions has been reported to occur in 2% of patient with giant cell tumour1.    

          There are approximately 50 cases reported of giant cell tumour with pulmonary metastases though only a few presented with symptoms from pulmonary lesions. One patient was treated with radiotherapy (3098 rads) for his pulmonary lesions and resection of primary tumour2, and another patient was treated with surgical excision of pulmonary nodules with hemipelvectomy for his primary lesion3.  

            There are very few large studies done on this topic. A study by Raphael et al4 in 1970 on 218 patients with giant cell tumour, reported the presence of pulmonary lesions in six patients, all appeared after the resection of primary lesion. They were treated with lobectomy and 5 out of 6 cases were successful. Another study by Robert et al in 19945 suggested that the overall incidence of Spread in giant cell tumour is 0-7%.                                                                                                          

        There is so far no agreement on how to treat patients with pulmonary metastasis and every patient is treated differently. Recently much emphasis has been on the use of interferon-alpha in optimizing pulmonary lesions from giant cell tumour1.

        The literature so far suggests that the patient presented with pulmonary lesions treated with radiotherapy showed inconsistent results with higher risk for sarcoma transformation. Another patient who underwent surgery had incomplete resection due to large number of lesions.                                                                               

        Our protocol to treat this patient initially with cytotoxic agents, en-bloc resection of primary site and to treat recurrence with surgical excision proved to be effective although, larger Studies are required to Support this. Our patient is now 29 Years old and has no problems. 

Reference : 

1. Yasko A.W: Giant cell tumour of bone. Curr Oncol Rep 2002 Nov; 4(6)520-61. 

           2. Cheng.J.C, Johnston.J.O: Giant cell tumour of bone, prognosis and treatment of pulmonary metastasis. Clin orthop.1997 ;( 338):205 – 214. 

           3. Bertoni F, Present.D and Enneking.W.F: Giant cell tumour of bone with pulmonary metastasis,JBJS 1985; 67-A: 890-900

           4.Goldenberg Raphael R., Campbell.C.J, and Bonfiglio.M: Giant cell tumour of bone, an analysis of 218 patients.JBJS 1970; 52-A (4):619-664      

          5. Kay.R.M, Eckardt.J.J, Seeger L.L, Mirra J.M and Hak.D.J: Pulmonary metastasis of benign giant cell tumour of bone, report of six cases.  Clin orthop.1994 ;( 302):219-230      

 

This is a peer reviewed paper 

Please cite as : Yasir Khan: Metastatic Navicular Giant Cell Tumour (Osteoclastoma)- A Case Report With 16 Year Follow-Up

J.Orthopaedics 2007;4(4)e21

URL: http://www.jortho.org/2007/4/4/e21

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