Case Report

Postpartum Treatment of Metastatic Recurrent Giant Cell Tumor of Capitate Bone of Wrist

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Giant cell tumors (GCTs) are aggressive benign lesions that occur in the bone epiphysis. They are most often found in the long bones of the lower extremities. Wrist bone involvement is rare, capitate bone involvement exceedingly rare. Treatment in the wrist usually consists of excision, local adjuvant treatment, and cementing and/or bone grafting. GCTs also metastasize to the lungs; pulmonary lesions are excised and systemic therapy provided.

We present the case of a 19-year-old woman with GCT of the capitate bone. After initial excision and cementing, the GCT recurred with lung metastases during a pregnancy. Rapid expansion of the GCT during pregnancy was reversed with systemic treatment with denosumab after pregnancy. Excellent response to this chemotherapy permitted limb-sparing surgery and wrist reconstruction with structural cortical autogenous bone graft. Resection of pulmonary metastases after a year of treatment with denosumab revealed lung parenchyma with calcification and ossification and limited viable tumor.


 

References

Take-Home Points

  • GCT of bones of the wrist is rare. This article is the only report of a wrist GCT during pregnancy that we could identify.
  • Routine treatment usually consists of surgical excision with local adjuvant, and in the wrist, often results in reduced wrist motion.
  • GCT of the wrist is more aggressive than the more common locations in long bones, with higher local recurrence rates if treated with surgery alone.
  • Diagnosis is often delayed for GCT of the wrist, due to insufficient imaging, which should include CT or MRI.
  • For pregnant women with GCT, local adjuvant treatments can be used in addition to surgery. Following pregnancy, denosumab can be used systemically, and can be effective with metastatic or unresectable disease.

Giant cell tumor (GCT) of bone accounts for about 5% of primary bone tumors. 1-3 Only 3% to 5% of GCTs occur in the hand. 4,5 Wrist involvement, which is rare, most often involves the hamate bone. 5-7 Capitate bone involvement is exceedingly rare. 8-11 Although histologically benign, GCT can recur locally after treatment with curettage alone, and lung metastases are found in 2% to 5% of cases. 2,12-14 Therefore, en bloc tumor excision is preferred in the setting of cortical erosion or soft-tissue involvement. 1,4,8 Wrist joint motion is inevitably reduced, and bone graft donor-site morbidity is significant. 6-8

In the unusual case reported here, GCT presented in the capitate bone and, after the patient became pregnant, recurred in the hamate and trapezoid bones with soft-tissue extension and lung metastases. The capitate was excised en bloc and reconstructed with an interposition of polymethylmethacrylate bone cement. Pulmonary metastases developed, and the GCT expanded to involve multiple carpal bones and the bases of the second through fourth metacarpals. A 10-month course of systemic chemotherapy with the RANK ligand (RANKL) inhibitor denosumab was started after the pregnancy. After this treatment, the patient underwent both tumor resection and reconstruction with autogenous bicortical iliac crest bone graft (ICBG) carefully designed to preserve range of motion and maintain the fingers in anatomical position. Treatment with denosumab was continued after surgery. Although this case offers no endpoint for postoperative chemotherapy with denosumab, preoperative treatment dramatically reduced the GCT and permitted limb-sparing reconstruction. The patient provided written informed consent for print and electronic publication of this case report.

Case Report

A 19-year-old right-handed woman with atraumatic swelling of the left wrist presented to an orthopedic surgeon at an outside facility. Physical examination revealed tender fullness on the dorsum of the wrist, slightly reduced range of motion and grip strength, and a neurovascularly intact wrist. The diagnosis was periarticular cyst, and the patient underwent physical therapy. Two years later, the swelling returned, tenderness was increasing, and symptoms did not resolve with cast immobilization. A radiograph showed a lytic lesion in the capitate bone ( Figure 1 ).

Figure 1.

GCT was diagnosed with percutaneous needle biopsy. A preoperative chest radiograph was reported normal. For initial treatment, the capitate and trapezoid bones were resected en bloc through a dorsal approach. Reconstruction consisted of limited arthrodesis using bone cement without additional fixation.

At 6-month follow-up, the patient was pregnant, and there was a recurrence of the wrist lesion. During the first 2 months of pregnancy, swelling and pain rapidly progressed, and a palpable mass formed. Radiographs showed a lytic lesion extending into the hamate bone ( Figure 2 ), and magnetic resonance imaging (MRI) showed articular extension of the lesion with involvement of the base of the fourth metacarpal.

Figure 2.
Targeted anti-RANKL therapy was not recommended (and was not available at the patient’s home hospital). The patient deferred surgical treatment because of the pregnancy, which proved otherwise uneventful and ended with a full-term delivery.

After the pregnancy, radiographs of the wrist showed complete destruction of the hamate and trapezium bones, with erosion of the bases of the second through fourth metacarpals ( Figure 3A ).

Figure 3.
The patient presented at our institution 4 years after initial diagnosis. Computed tomography (CT) of the chest showed numerous bilateral pulmonary nodular opacities. Wrist imaging showed soft-tissue extension ( Figure 3B ). The diagnosis of recurrent metastatic GCT was confirmed with needle biopsies of the wrist mass and the right lung nodule.

Systemic chemotherapy was initiated with 120 mg of denosumab, given subcutaneously on days 1, 8, and 15 and then monthly during the 10 months leading up to surgery. Serum calcium was monitored during treatment and remained within the normal range the entire time, except for once at the start of

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