GIANT CELL TUMOURS
OF T H E H A N D
B. K. S. S A N J A Y , G. A. R A J a n d D . A. Y O U N G E
From the Central Institute of Orthopaedics, Safdarjang Hospital New Delhi, India and the Department of Orthopaedic Surgery, King Faisal Specialist Hospital, Riyadh, Saudi Arabia A study of seven cases of giant cell tumours of the hand is reported. All tumours were treated by an en bloc excision of the tumour or by whole ray resection. En bloc resection of the tumour and reconstruction with a fibular graft where necessary should be considered as the treatment of choice in giant cell turnout of the hand.
Journal of Hancl Surgery (British and European Volume, 1996) 21B: 5." 683-687 Giant cell tumour of bone accounts for 5% of all primary bone tumours in the West (Dahlin & Unni, 1986; Goldenberg et al, 1970; Jaffe, 1953; Jaffe et al, 1940; McGrath, 1972). However, this tumour is the most common primary bone tumour in China and India, accounting for 20% in the former (Sung et al, 1982) and 30% in the latter (Reddy et al, 1974). Giant cell tumours in the hand are rare. Minguella (1982) found that only 3% of all giant cell tumours of bone affected the hand. Averill et al (1980) reviewed 20 reported series of 1,228 giant cell tumours of bones. They found that 2% occurred in hand bones, almost 1% in metacarpals and 1% in phalanges. Although giant cell tumours of bone are four to six times more common in India and China, giant cell turnouts of the hand are not common. Series from India and China report 2.5% in the metacarpal (Gupta et al, 1980; Sanjay et al, 1989) and 2.2% in the phalanges (Reddy et al, 1974; Rockwell and Small, 1961; Sung et al, 1982; Tuli et al, 1978).
MATERIALS A N D M E T H O D S
The clinical data of seven giant cell tumours of the hand bones are given in Table 1. Two of them have been reported earlier (Sanjay et al, 1989; Sanjay and Younge, 1996). There were three male and four female patients with an average age of 24.5 years (range 14-35 years). Patients presented with a gradually increasing swelling, with or without pain. Average duration of symptoms in all but one patient was 6.3 months, with a range of 4 to 8 months. The tumour presented as an expansile lesion destroying the cortex in six cases. All of them had extension of the tumour into the soft tissues. In one case, the tumour involved two phalanges of one finger. Chest X-rays were negative for metastases in all except one patient, who presented with multiple nodules in both lungs. Before definitive surgery, the pathological diagnosis was made by fine needle aspiration cytology in four cases and by open biopsy in three. Gross examination showed that the cortex was broken in six cases. The average size was 7 x 3 x 4 cm. Tumours con-
Table 1--Clinical data of giant cell tumours of hand
Case
Age (in years)/sex
Site of tumour
Size of tumour (in cm)
Duration of symptoms
Treatment
Follow-up (in years)
Local recurrence
En bloc t u m o u r excision and fibular graft En bloc t u m o u r excision and fibular graft En bloc t u m o u r excision and fibular graft En bloc ray resection and medial transposition of index ray En bloc ray resection En bloc ray resection 1. En bloc ray resection 2. Excision of soft tissue recurrence
14
No
11
No
9
No
7
No
13
No
7
No
1
35/F
Left 1st metacarpal
6x 5×6
4 months
2*
14/F
Left 3rd metacarpal
6x 5x 5
6 months
3
25/M
Right 1st metacarpal
5x4x4
6 months
4
32/M
Right middle and proximal phalanges
7x4x4
8 months
5
25/M
3x3x2
6 months
6
24/F
4x4x3
8 months
7*
17/F
Right 2nd metacarpal Left 5th metacarpal Left 5th metacarpal
6x 5x 3
3.5 years
* Reported earlier (Sanjay et al, 1989; Sanjay and Younge, 1996). 683
15
Remarks
No recurrence after Pulmonary and second surgery skeletal metastases
684 sisted of soft grayish tissue with areas of haemorrhage. The periphery of tumours cut easily with a knife. All tumours were treated by an en bloc excision of the tumour or the whole ray. Tumours of the thumbs (Figs 1 and 2) and long finger metacarpals (Fig 3) were treated with en bloc excision of the tumour followed by reconstruction with a fibular graft (three cases). Tumour of the phalanges of the middle finger (Fig 4) was treated by ray resection and medial transposition of the index finger. Tumours of index (Fig 5) and small finger metacarpals (Fig 6) were treated by ray resection only (three cases). The fibula is twice as thick as any metacarpal, therefore, the fibula was split longitudinally in replacing the long finger metacarpal to get the required thickness. Fibular grafts and the transposed ray were fixed with K-wires, which were removed 6 to 12 weeks after surgery. RESULTS Follow-up ranged from 7 to 15 years (average 11 years; Table 1). Six patients had their first operation done by us and none of the tumours recurred. One patient (Case 7) had the first operation in the referring hospital. Three months later, she developed soft tissue recurrence. Chest X-ray taken at this time showed multiple nodules in both lungs (Fig 7). Chest X-ray taken at the first presentation did not show any evidence of metastases. At this stage, the patient was referred to us for treatment of the soft tissue recurrence and lung metastases. The nodule on the left hand was excised. The lung metastases were treated by chemotherapy. Pulmonary metastases had been growing during and after chemotherapy for 2 years, but stopped growing 8 years after the initial diagnosis of the tumour. The
THEJOURNALOF HANDSURGERYVOL.21BNo. 5 OCTOBER1996 chest X-ray taken 14 years after diagnosis showed that all large nodules had disappeared (Fig 8). A skeletal survey and bone scan taken 7 years after diagnosis revealed the presence of multiple bone metastases. The latest bone scan shows persistence of bone metastases. The patient remained asymptomatic since the first presentation of metastases. DISCUSSION More than 95% of cases of recurrence of giant cell tumour of bone occur within 2 years of surgery (Goldenberg et al, 1970) and in the hand, tumours occur within a year (Averill et al, 1980). Recurrence within a few months of surgery should usually be considered, caused by inadequate surgery, such as simple curettage. Primary giant cell tumours of long bones rarely present with all-round cortical destruction of the bone and soft tissue extension of tumour. However, giant cell tumours of hand bones usually present as an expansile lesion with a paper-thin cortex, pathological fracture and soft tissue extension. These features make curettage more difficult and a less effective treatment. Curettage alone or curettage with bone grafting is usually not effective treatment, even in giant cell tumours of the long bones (Goldenberg et al, 1970; Hutter et al, 1970; McDonald et al, 1986; McGrath, 1970; Sanerkin, 1962; Sung et al, 1982), and it becomes less effective in treating tumours in the hand (Averill et al, 1980; McGeoch and Varian, 1985). In general, the best results have been obtained by en bloc excision of giant cell tumours (Goldenberg et al, 1970; McDonald et al, 1986; McGrath, 1972; Sung et al, 1982) and this also applies in the hand (Averill et al,
b Fig 1 Case 1. (a) Preoperative X-rays. (b) Postoperative X-ray showing replacement with a fibular graft. The head of the fibula liesproximallyand looks like the base of the metacarpal.
Fig 2
Case 1. (a) Clinical photograph at presentation showing globular swellingextendingfrom the wristjoint to the metacarpal phalangealjoint. (b) 2 years after operation.
GIANT CELL TUMOURS
685
Fig 5 Fig 3
(b) Tumour was treated with 2nd ray resectionat the base of the second metacarpal.
Case 2. (a) Preoperative X-ray of the hand showing a radiolu-
cent expansile lesion involving the entire diaphysis of the metacarpal. (b) PostoperativeX-ray. Reproducedfrom Sanjay BKS et al (1989).
Fig 4
Case 5. (a) X-ray of hand of Case 5 showing osteolytic lesion in the distal half of the index finger metacarpal. The cortex was broken and there was associated soft tissue swelling.
Case 4. (a and b) X-ray showing destruction of the whole of the middle and the distal half of the proximal phalanx of the middle finger with soft tissue involvement. (c) Postoperative X-ray.
1980; Besser et al, 1987; Gupta, 1980; Patel et al, 1987; Shaw and Mosher, 1983). Reconstruction with an autograft should be done where necessary to obtain a functional and a cosmetically acceptable hand (McGeoch and Varian,,1985; Minguella, 1982; Sanjay et al, 1989). The incidence of pulmonary metastases from benign giant cell tumours in different series has varied from 0 to 2% (Bertoni et al, 1985; 1988; Campanacci et al, 1987; Rock et al, 1984). Metastatic lesions are histologically indistinguishable from the primary lesion (Bertoni et al, 1988; Campanacci et al, 1987; Dahlin and Unni, 1986; Goldenberg et al, 1970). Several authors have noted that multiple operative procedures on the primary tumour seem to promote pulmonary metastasis (Bertoni et al, 1988; Jaffe et al, 1940). However, cases have been reported with radiological evidence of pulmonary
Fig 6
Case 6. (a) Preoperative X-ray of the hand showing the
expansile lytic tumour of the small finger metacarpal. The cortex was broken and tumour extended into the soft tissues. (b) Specimenof whole ray after resection. metastases before any surgery (Bertoni et al, 1988; Goldenberg et al, 1970; Sanjay et al, 1993). Giant cell tumour in the hands has been considered to be aggressive, with an earlier and higher rate of recurrence than in other skeletal sites (Patel et al, 1987). In a study of 21 patients of giant cell tumour of the hand, two patients (5%) developed pulmonary metastases (Averill et al, 1980). It seems that giant cell tumour of bones in the hand is not only aggressive and associated with a higher incidence of recurrence but also has a predilection for pulmonary metastases. Most of the metastases have been to the lung. Metastasis to bone is
THE JOURNAL OF HAND SURGERY VOL. 21B No. 5 OCTOBER 1996
686
presence of metastatic nodules (Bertoni et al, 1988). This favourable prognosis may be independent of the type of treatment used. References
Fig 7
Case 7. C h e s t X - r a y t a k e n at the time of first p r e s e n t a t i o n to us, s h o w i n g m u l t i p l e n o d u l e s in b o t h lungs. R e p r o d u c e d f r o m Sanjay and Y o u n g e (1996).
Fig 8
C h e s t X - r a y t a k e n 14 years after diagnosis, s h o w i n g t h a t all nodules have disappeared. Reproduced from Sanjay and Y o u n g e (1996).
extremely rare. A review of the literature revealed only three cases (Bertoni et al, 1985; 1988; Lasser and Tetewsky, 1957; Rock et al, 1984; Vanel et al, 1983). As with the primary lesion, the natural history and the clinical behavior of metastatic giant cell tumour is unpredictable, and the time lapse of metastasis has varied in different studies, averaging between 16 months (Bertoni et al, 1985) and 5 years (Rock et al, 1984). Metastases have been noted at the time of first presentation in other studies (Goldenberg et al, 1970; Sanjay et al, 1993). The results of surgery, chemotherapy and radiotherapy have varied in different series. Some metastases have been asymptomatic for long periods without any treatment, and in some cases metastases have regressed without treatment. In others, existing nodules have progressed and additional nodules appeared, and some nodules regressed and others progressed even during and after chemotherapy (Sanjay and Younge, 1996). Death may occur irrespective of any treatment including pulmonary resection, chemotherapy and radiotherapy. Another review on giant cell tumours with pulmonary metastases indicates that 85% of patients survived in the
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687 Received: 21 December 1995 Accepted after revision: I April 1996 Dr B. K. S. Sanjay, Deparlanent of Orthopaedic Surgery, MBC 77, King Faisal Specialist Hospital and Research Centre, PO Box 3354, Riyadh 11211, Saudi Arabia. © 1996 The British Society for Surgery of the Hand