SPINE

Cervicothoracic Giant Cell Tumor Expanding Into the Superior Mediastinum: Total Excision by Combined Anterior-Posterior Approach

By Katsuhito Yoshioka, MD; Norio Kawahara, MD; Hideki Murakami, MD; Satoru Demura, MD; Masahito Kawaguchi, MD; Makoto Oda, MD; Isao Matsumoto, MD; Katsuro Tomita, MD
ORTHOPEDICS 2009; 32:531

Abstract

This article describes a case of cervicothoracic giant cell tumor expanding into the superior mediastinum treated by total spondylectomy. A 42-year-old-man presented with back pain and paraparesis. Magnetic resonance imaging revealed the collapse of the T2 vertebral body. The spinal cord was severely compressed by the tumor mass. The tumor had spread from T2 to the mediastinum, so that the tumor was in contact with many vital structures. To resect the tumor completely, total spondylectomy from T1 to T3 was performed through a combined anterior-posterior approach. The tumor was dissected from the vital structures using an anterior low cervical approach and splitting one-third of the sternum. En bloc vertebral resection from Th1 to Th3, including the tumor pseudocapsule, was possible through a posterior approach. The tumor around the nerve roots or dura was resected piece by piece since it was possible to separate the capsulated tumor from the dura. Splitting one-third of the sternum allowed separation of the tumor from the anterior vital structures, under direct vision. This allowed en bloc vertebral resection of the tumor that had spread to the mediastinum from T2 and in the craniocaudal direction from T1 to T3. Although giant cell tumor is benign, it can be locally aggressive. Complete excision of a giant cell tumor is the best treatment option even for the cervicothoracic spine, to protect the vital structures or neural function.

Spinal involvement of giant cell tumor is relatively rare,1,2 and there are few reports related to it arising in the cervicothoracic vertebra.3-5 Spinal giant cell tumor can be locally aggressive, and curettage leads to a high rate of local recurrence.6 It is generally agreed that complete excision of a giant cell tumor is the best treatment option; however, a complete excision is often not a feasible option for a giant cell tumor arising in the cervicothoracic spine, especially if the tumor has expanded into the superior mediastinum, because of the presence of major blood vessels and the vagus nerve, trachea, and esophagus.

This article describes case of giant cell tumor that underwent complete excision. The tumor had originated in T2 and expanded into the 2 adjacent vertebrae and the superior mediastinum.

Case Report

A 42-year-old man presented with a 7-month history of back pain. He reported bilateral leg weakness and numbness 6 months after the first onset of pain. His paraparesis gradually worsened so that on admission he was unable to stand.

Radiographs showed a radiolucent lesion at T2 and the absence of the bilateral pedicle shadow. Computed tomography (CT) scans revealed the collapse of the T2 vertebral body and the destruction of the vertebral cortex, pedicle, lamina, and posterior aspect of T1 and T3 vertebral bodies (Figure 1). Magnetic resonance imaging (MRI) showed that the spinal cord was severely compressed by the tumor mass. It had invaded the spinal canal from the T2 lamina and had advanced through the posterior wall of the vertebral body. The tumor had spread to the mediastinum from T2 and was in contact with many vital structures including the trachea, esophagus, carotid artery, subclavian artery, and aortic arch (Figure 2). Further investigation revealed no other tumor lesions. A needle biopsy was performed, and the histological diagnosis was giant cell tumor. To resect this tumor completely, a combined anterior-posterior approach was planned. The patient underwent total spondylectomy from T1 to T3.

Figure 1A: Preoperative midsagittal CT image Figure 1B: Preoperative axial CT images

Figure 1: Preoperative midsagittal (A) and axial (B) CT images. The T2 vertebral body was collapsed (arrow). CT revealed destruction of the vertebral cortex, pedicle, lamina, and posterior aspect of T1 and T3 vertebral bodies.
Figure 2A: The spinal cord was severely compressed by the tumor mass Figure 2B: The tumor had spread to the mediastinum from T2 Figure 2C: The tumor was in contact with many vital structures

Figure 2: Preoperative axial at T2 (A), midsagittal (B), and left sagittal (C) MRIs. The spinal cord was severely compressed by the tumor mass. The tumor (arrows) had spread to the mediastinum from T2 and was in contact with many vital structures including the trachea, esophagus, carotid artery, subclavian artery, and aortic arch.

To separate the tumor from anterior vital structures, an anterior approach was performed under direct vision. Based on a conventional anterior cervical approach, an incision was made on the left side along the anterior border of the sternocleidomastoid and over the midline of the sternum. The upper one-third of the sternum was split longitudinally to expose the T3 level. The esophagus, trachea, brachiocephalic artery, and left recurrent laryngeal nerve were gently retracted to the right side. The left common carotid artery, left subclavian artery, and vagus nerve were retracted to the left side. The thoracic duct was identified and carefully ligated. The left anterolateral side of the tumor was dissected from these structures. Next, the esophagus and the trachea were retracted to the left side and the brachiocephalic trunk was retracted to the right side, and the tumor edge on the right side was exposed and dissected. Splitting one-third of the sternum allowed the T3 to T4 region to be visible. The tumor was then separated from the aortic arch. Because of severe adhesion between the tumor and parietal pleura, partial resection of parietal pleura was performed to achieve a sufficient safety margin.

The patient was then placed in the prone position. The posterior elements between C5 and T6 were widely exposed. The tumor had invaded the T2 lamina and compressed the spinal cord. After insertion of pedicle screws at C5, C6, T5, and T6, a laminectomy from C7 to T4 was performed in piecemeal fashion. Bilateral T2, T3, and left T1 nerve roots involved by the tumor were atrophic and were cut, but bilateral C8 and right T1 nerve roots and dura were carefully detached from the capsulated tumor in order to preserve neurological function. The tumor around the dura was then thoroughly resected piece by piece. En bloc excision of the T1, T2, and T3 vertebral bodies was carried out after cutting the inferior border of C7 and the superior border of T4 using a diamond T-saw (Medtronic Sofamor Danek, Inc, Memphis, Tennessee). The vertebral body was then reconstructed using a titanium mesh cage filled with autogenous iliac crest.

Operative time was 1017 minutes with a blood loss of 4000 mL. There were no perioperative complications. Radiotherapy was not performed. Histological examination of the resected specimens of T2 and T3 vertebral bodies expanding to the superior mediastinum revealed negative margins at the proximal, distal, and anterior sites (Figure 3).

Figure 3A: Macroscopic photographs of the resected specimen Figure 3B: Macroscopic photographs of the resected specimen

Figure 3: AP (A) and axial (B) macroscopic photographs of the resected specimen. Histological examination revealed negative margins at the anterior site (C). The tumor was covered with capsule.

The patient, who had had preoperative incomplete paralysis (Frankel C), had regained a Frankel E ambulatory status 3 months postoperatively. Despite the fact that the atrophied left T1 nerve root was sacrificed, his grip strength had decreased little. This was likely due to compensatory mechanisms contributing to preserving neurological function. Radiography, CT scan, and MRI 2 years postoperatively showed no sign of tumor recurrence (Figure 4). The patient returned to working full-time.

Figure 4A: AP and lateral radiographs and midsagittal CT scan 2 years postoperatively. Figure 4B: AP and lateral radiographs and midsagittal CT scan 2 years postoperatively. Figure 4C: Midsagittal CT scan 2 years postoperatively.

Figure 4: AP (A) and lateral radiographs (B) and midsagittal CT scan (C) 2 years postoperatively.

Discussion

Giant cell tumor is histologically benign, but its clinical behavior is complex. Local recurrence rate after curettage is 26% to 50%.7 Campanacci et al2 reported a recurrence rate of 27% after intralesional curettage vs rates of 8% and 0% after marginal or wide resection, respectively. Options for adjuvant treatment for patients with giant cell tumor include cement implantation, cryosurgery, and phenolization when intralesional curettage was done in the limbs.

In the spine, because of the presence of major blood vessels and the spinal cord, these adjuvant treatments are risky. The use of radiation therapy remains controversial. Generally, radiotherapy for giant cell tumors of bone is discussed as a postoperative treatment in cases of nonradical tumor excision, or as a palliative treatment for postoperative recurrences or unresectable large tumors.8-10 Some authors do not recommend it because of the risk of sarcomatous change that occurs in 18.2% to 20% of patients.2,11,12 In addition, radiation myelopathy is a devastating complication of clinical radiotherapy.13 Junming et al3 reported that local recurrence was detected in 5 of 7 cases (71.4%) after subtotal resection, in spite of a combination with radiotherapy. Therefore, many authors advocate total en bloc excision or complete excision including tumor margins during the initial surgery for giant cell tumor in the spine to prevent recurrence.1,7,14-19

Few descriptions exist of complete excision of giant cell tumor with total spondylectomy (even intralesional) without adjuvant therapy.7,15 Hart et al7 reported 14 of 16 cases with no postsurgical recurrence in cases of total spondylectomy without adjuvant therapy. Tomita et al20 reported a surgical strategy for primary spinal tumors. They reported that total tumor excision including the tumor capsule, in en bloc or piecemeal fashion, is mandatory for benign tumors such as the giant cell tumor. Murata et al19 described tumor excision including tumor margin to treat tumor cells invading the anterior or posterior ligament tissue through the vertebral cortex.

Because the anatomy is relatively complicated, surgical treatment is not easy for tumors in the cervicothoracic spine. Furthermore, when the tumor has extended to adjacent verebra and superior mediastinum, as in our case, complete excision is difficult. Postoperative tumor recurrence at the superior mediastinum is a serious complication, the tumor should be completely removed, even a large tumor that has expanded to the superior mediastinum. Accordingly, we consider en bloc excision to be a primary option when possible. In the first step, to maintain the adequate tumor margin, we dissected the tumor from the vital structures including the trachea, esophagus, vagus nerve, carotid artery, subclavian artery, and aortic arch using an anterior approach. This procedure helped en bloc vertebral resection through a posterior approach to remove the whole tumor mass including tumor pseudocapsule.

However, the tumor around the nerve roots or dura was resected piece by piece. We had no choice but to cut bilateral T2, T3, and left T1 nerve roots involved by the tumor, but dura was preserved intact because it was possible to separate capsulated tumor from the dura. As a consequence, total excision including tumor margin was possible.

A follow-up period of 2 years is relatively short, so we continue following up regarding tumor recurrence.

References

  1. Dahlin DC. Giant-cell tumor of vertebrae above the sacrum: a review of 31 cases. Cancer. 1977; 39(3):1350-1356.
  2. Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am. 1987; 69(1):106-114.
  3. Junming M, Cheng Y, Dong C, et al. Giant cell tumor of the cervical spine: a series of 22 cases and outcomes. Spine. 2008; 33(3):280-288.
  4. Schwimer SR, Bassett LW, Mancuso AA, Mirra JM, Dawson EG. Giant cell tumor of the cervicothoracic spine. AJR Am J Roentgenol. 1981; 136(1):63-67.
  5. Shoda N, Nakajima S, Seichi A, et al. Computer-assisted anterior spinal surgery for a case of recurrent giant cell tumor. J Orthop Sci. 2002; 7(3):392-396.
  6. Balke M, Schremper L, Gebert C, et al. Giant cell tumor of bone: treatment and outcome of 214 cases. J Cancer Res Clin Oncol. 2008; 134(9):969-978.
  7. Hart RA, Boriani S, Biagini R, Currier B, Weinstein JN. A system for surgical staging and management of spine tumors. A clinical outcome study of giant cell tumors of the spine. Spine. 1997; 22(15):1773-1782.
  8. Miszczyk L, Wydma?ski J, Spindel J. Efficacy of radiotherapy for giant cell tumor of bone: given either postoperatively or as sole treatment. Int J Radiat Oncol Biol Phys. 2001; 49(5):1239-1242.
  9. Chakravarti A, Spiro IJ, Hug EB, Mankin HJ, Efird JT, Suit HD. Megavoltage radiation therapy for axial and inoperable giant-cell tumor of bone. J Bone Joint Surg Am. 1999; 81(11):1566-1573.
  10. Caudell JJ, Ballo MT, Zagars GK, et al. Radiotherapy in the management of giant cell tumor of bone. Int J Radiat Oncol Biol Phys. 2003; 57(1):158-165.
  11. Cahan WG, Woodard HQ, Higinbotham NL, Stewart FW, Coley BL. Sarcoma arising in irradiated bone; report of 11 cases. Cancer. 1948; 1(1):3-29.
  12. Berman HL. The treatment of benign giant-cell tumors of the vertebrae by irradiation. Radiology. 1964; (83):202-207.
  13. Berger JR. Medical mylopathies. In: Herkowitz HN, Garfin SR, Balderston RA, Eismont FJ, Bell GR, Wiesel SW, eds. The Spine. 4th ed. Philadelphia, PA: Saunders; 1999:1413-1430.
  14. Ido K, Shimizu K, Matsuda Y, et al. Total en bloc spondylectomy for giant cell tumor in the thoracic spine: a case report. Neuro Orthopedics. 1997; (21):75-80.
  15. Sanjay BK, Sim FH, Unni KK, McLeod RA, Klassen RA. Giant-cell tumours of the spine. J Bone Joint Surg Br. 1993; 75(1):148-154.
  16. Shikata J, Yamamuro T, Shimizu K, Shimizu K, Kotoura Y. Surgical treatment of giant-cell tumors of the spine. Clin Orthop Relat Res. 1992; (278):29-36.
  17. Tomita K, Kawahara N, Baba H, Tsuchiya H, Fujita T, Toribatake Y. Total en bloc spondylectomy. A new surgical technique for primary malignant vertebral tumors. Spine. 1997; 22(3):324-333.
  18. Stener B, Johnsen OE. Complete removal of three vertebrae for giant-cell tumour. J Bone Joint Surg Br. 1971; 53(2):278-287.
  19. Murata A, Fujita T, Kawahara N, et al. Giant cell tumor of the spine. Histological analysis of giant cell tumor of the spine treated with total en bloc spondylectomy [in Japanese]. Rinsho Seikei Geka. 2000; (35):723-728.
  20. Tomita K, Kawahara N, Murakami H, Demura S. Total en bloc spondylectomy for spinal tumors: improvement of the technique and its associated basic background. J Orthop Sci. 2006; 11(1):3-12.

Authors

Drs Yoshioka, Kawahara, Murakami, Demura, Kawaguchi, and Tomita are from the Department of Orthopedic Surgery, and Drs Oda and Matsumoto are from the Department of General and Cardiothoracic Surgery, Kanazawa University, Kanazawa, Japan.

Drs Yoshioka, Kawahara, Murakami, Demura, Kawaguchi, Oda, Matsumoto, and Tomita have no relevant financial relationships to disclose.

Correspondence should be addressed to: Katsuhito Yoshioka, MD, Department of Orthopedic Surgery, School of Medicine, Kanazawa University, 13-1 Takaramachi, Kanazawa, 920-864 1 Japan.

DOI: 10.3928/01477447-20090527-26

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