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abstractFull article available online at ORTHOSuperSite.com. Search: 20120222-42

This article describes a case of a giant cell tumor that expanded into the thoracic cavity and through the spinal canal into the vertebrae. A 36-year-old man presented with a 6-month history of back pain and dyspnea. Plain chest radiographs showed a huge mass accompanied by right pleural effusion. The mass involved the 12th thoracic spine, and the spinal cord was severely compressed. The tumor was resected with a 2-stage procedure. As a first stage to separate the tumor from the anterior vital struc-tures under direct vision, thoracic surgeons performed a right thoracotomy with chest wall reconstruction from the 8th to 11th ribs. The right lung and inferior vena cava were gently retracted, and the tumor was carefully detached from these structures. We were not able to separate the tumor from the right diaphragm due to severe invasion; therefore, we performed partial resection of the right diaphragm with the tumor. After excision of the anterior part of the tumor, the thoracic wall was reconstructed with the right eighth rib and Marlex mesh. When the patient’s general condition improved 2 weeks later, spondylectomy by posterior approach was performed. We achieved exci-sion of a giant cell tumor that had expanded into the thoracic cavity and through the spinal canal into the vertebrae. The patient had achieved full rehabilitation with no neurological or respiratory abnormalities at 7 years postoperatively.

Drs Demura, Kawahara, Murakami, Akamaru, Kato, Tomita, and Tsuchiya are from the Department of Orthopaedic Surgery, Dr Oda is from the Department of General and Cardiothoracic Surgery, and Dr Kawahara is also from the Department of Orthopaedic Surgery, Kanazawa University, Kanazawa, Japan.

Drs Demura, Kawahara, Murakami, Akamaru, Kato, Oda, Tomita, and Tsuchiya have no relevant financial relationships to disclose.

The authors thank Ryan Ilgenfritz, MD, and William C. Hutton, DSc, for their criticism and advice.Correspondence should be addressed to: Satoru Demura, MD, Department of Orthopaedic Surgery,

Kanazawa University, 13-1 Takaramachi, Kanazawa, 920-8641, Japan (demudon@med.kanazawa-u.ac.jp).doi: 10.3928/01477447-20120222-42

Giant Cell Tumor Expanded Into the Thoracic Cavity With Spinal InvolvementSatoru Demura, mD; Norio Kawahara, mD; hiDeKi muraKami, mD; tomoyuKi aKamaru, mD; SatoShi Kato, mD; maKoto oDa, mD; KatSuro tomita, mD; hiroyuKi tSuchiya, mD

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Figure: Photographs of the resected specimens from the thoracic cavity (A) and the vertebral body (B).

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n Case Report

Giant cell tumors are benign prima-ry bone tumors that usually occur in the epiphysis of the long bones

but rarely in the axial skeleton. They are found most commonly after skeletal ma-turity and show slight female predomi-nance.1-3 Histologically, giant cell tumors consist of multinucleated osteoclastic gi-ant cells in the stroma of spindle-shaped cells.4 Because giant cell tumors are typi-cal aggressive locally, curettage can lead to a high rate of local recurrence.1,5 In the extremities, it has been reported that the combination of adjuvant therapy reduces the rate of local recurrence.5-8 When the spine is involved, these adjuvant thera-pies present the risk of postoperative complications because of the presence of major blood vessels and the spinal cord. Therefore, complete excision of giant cell tumors is generally considered to be the best treatment option. However, due to the difficulties of removing spinal tumors, complete resection is often feasible for gi-ant cell tumors, especially when they have expanded into the thoracic cavity. This ar-ticle describes a case of a giant cell tumor that expanded into the thoracic cavity and had spinal involvement.

Case RepoRtA 36-year-old man presented with a

6-month history of back pain and dyspnea. A week before our workup, the patient had worsening dyspnea and presented at his primary hospital. After examination, he was referred to our institution due to an abnormal shadow on his chest radio-graphs. He reported dyspnea at rest, and physical examination showed diminished right breath sounds. An arterial blood gas revealed a pH of 7.41, carbion dioxide par-tial pressure (pCO2) of 46 mmHg, oxygen partial pressure (pO2) of 66 mmHg, and an oxygen saturation of 93% on room air. No neurological deficits existed in his lower limbs. Plain chest radiographs showed an atelectatic region in the right thoracic cav-ity (Figure 1). In addition, disappearance of the right 12th rib and absence of the right

pedicle shadow at T12 was observed.

Computed to-mography (CT) scan revealed a large mass in the right hemitho-rax accompanied by pleural effusion. Due to the mass, the right lung was se-verely compressed, and the mediasti-num was shifted to the left. The mass also involved the 12th thoracic ver-tebral body, right pedicle, and lamina (Figure 1). Coronal magnetic resonance imaging (MRI) re-vealed a 25312-cm mass that enhanced heterogeneously by gadolinium (Figure 2). Invasion of the tumor mass into the spinal canal through the vertebrae was also observed (Figure 2). Further investigation revealed that no other tumor lesions existed. Biopsy confirmed a giant cell tumor.

To resect the tumor, we performed a 2-stage procedure. As a first stage to sepa-rate the tumor from the anterior vital struc-tures under direct vision, a right thoracoto-my with chest wall reconstruction from the 8th to 11th ribs was performed by a tho-racic surgeon (M.O.). Adhesions existed between the tumor and the right lung, in-ferior vena cava, and right hemidiaphragm. The right lung and inferior vena cava were gently retracted, and the tumor was care-fully detached from these structures. We were not able to separate the tumor from the right diaphragm because of severe in-vasion; therefore, we performed partial resection of the right diaphragm with the

tumor. After excision of the anterior part of the tumor (Figure 3), the thoracic wall was reconstructed with the right eighth rib and Marlex mesh. Operative time was 460 min-utes, with an intraoperative blood loss of 8700 mL. Postoperative intensive respira-tory and circulatory care were given. After excision of the anterior part of the tumor, the arterial blood gas improved to a pO2 of 91 mmHg and oxygen saturation improved to 98% on room air.

When the patient’s general condition improved 2 weeks later, the second stage of the procedure, spondylectomy by poste-rior approach, was performed. After inser-tion of pedicle screws from T9 to L3, lami-nectomies at T11 and T12 were performed. The tumor compressed the dura matter, and thus was carefully detached from the tumor

Figure 1: Chest radiograph showing an atelectatic region in the right thoracic cavity (A). Reconstructed sagittal computed tomography showing the mass involved the vertebral body, pedicle, and posterior elements of T12 (B).

Figure 2: Coronal magnetic resonance image with gadolinium showing a 25312-cm mass in the thoracic cavity (A). Axial T2-weighted magnetic resonance image showing invasion of the tumor mass into the spinal canal through the vertebrae (B).

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to preserve neurological function. Total spondylectomy was performed after cut-ting the inferior border of the T12-L1 disk and the superior border of the T10-T11 disk using the T-saw (Kashiwa, Kanazawa, Japan) (Figure 3). The vertebral body was then reconstructed using a titanium mesh cage filled with autogenous iliac crest bone. Pathologic examination showed a gi-ant cell tumor with proliferation of multi-nucleate giant cells and mononuclear cells, with no significant cellular atypia.

One year postoperatively, the patient was found to have developed a subcutane-ous recurrence of the tumor at the surgical wound site, likely due to seeding of the site during a prior surgery. This new growth was successfully excised. Four years post-operatively, he underwent revision surgery with anterior reconstruction using a titani-um cage and fibula strut graft due to pseud-arthrosis. Seven years postoperatively, fol-low-up imaging showed no signs of tumor recurrence and complete bony fusion at the reconstruction site; 1 of the pedicle screws at right T9 was seen to be placed laterally (Figure 4). The patient achieved full reha-bilitation with no neurological or respira-tory abnormalities.

DisCussionGiant cell tumors are histologically be-

nign tumors; however, they often take an aggressive course clinically. Local recur-rence rates after intralesional curettage have been reported to be high, and some options exist for adjuvant treatment, in-cluding polymethylmethacrylate implan-tation,7 cryosurgery,8 and phenolization in the extremities.6 However, in the spine, physical adjuvant treatments carry the risk of damaging the spinal cord, nerve roots, and major blood vessels. Therefore, thor-ough excision during the initial surgery is the best course of action for giant cell tu-mors in the spine to prevent recurrence.5,9-11

Few cases involving giant cell tumors occupying the thoracic cavity have been reported in the literature.12,13 The tumor in our patient was not detected until it

had expanded into the thoracic cavity. Even if a large tu-mor is formed and lung involvement or compression occurs, it can be difficult to detect the tumor in the thoracic cavity due to its mild sub-jective symptoms. Furthermore, inva-sion of the tumor into the vertebral body, pedicle, and lamina made com-plete excision of the tumor difficult. Some reports rec-ommended that sur-gical resection fol-lowed by radiother-apy may be effective because complete excision of giant cell tumors in ver-tebrae is difficult.2,13

However, several authors recommend against radiotherapy because of the risk of sarcomatous change, which occurs in 27% of patients.1 Therefore, radiotherapy for giant cell tumors might be limited as a postoperative treatment to cases of pal-liative treatment of postoperative recur-rences or unresectable large tumors.

As our first-stage procedure, we re-moved the tumor from the vital structures (including the lung and inferior vena cava) by anterior approach to improve respira-tory and circulatory functions. A thoracic surgeon should be consulted to dissect the lung from the tumor capsule and perform chest wall reconstruction. After stabiliz-ing the patient’s general condition, we performed total spondylectomy without damaging the spinal cord.

En bloc excision is feasible for aggres-sive tumors such as giant cell tumors. In a review of aggressive benign tumors, in-cluding giant cell tumors, Harrop et al14

recommended en bloc resection in the thoracic and lumbar spine. We excised the tumor in 2 stages. The patient had a subcu-taneous recurrence in the surgical wound 1 year postoperatively, possibly because of intraoperative contamination. However, no recurrence occurred at the primary site, and no evidence of disease existed for 6 years. This good result was probably due to capsule excision of the tumor. In addi-tion, giant cell tumors are not malignant; therefore, preserving neurological func-tion could be considered a higher priority. Junming et al15 discussed 22 cases of cer-vical giant cell tumors, in which local re-currence was detected in 5 of 7 cases that underwent subtotal resection. However, total excision was performed in 1 of 13 cases (even intralesional).15 Tomita et al11 considered it mandatory to perform total tumor excision, including the tumor cap-sule, in an en bloc or piecemeal fashion for benign tumors such as giant cell tu-

Figure 3: Photographs of the resected specimens from the thoracic cavity (A) and the vertebral body (B).

Figure 4: Chest radiograph 7 years postoperatively showing an atelectatic region in the right thoracic cavity almost recovered (A). Lateral radiograph of the thoracic spine (B). Reconstructed sagittal computed tomography scan showing complete bony fusion at the reconstruction site (C).

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n Case Report

mors. Total excision of tumors, including the capsule, should be performed en bloc or intralesionally.

We achieved excision of a giant cell tu-mor that had expanded into the thoracic cav-ity and through the spinal canal into the ver-tebrae. The patient had achieved full reha-bilitation with no neurological or respiratory abnormalities at 7 years postoperatively.

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