Extensive total spondylectomy for recurrent giant cell tumor in the thoracic spine

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1 J Neurosurg Spine 6: , 2007 Extensive total spondylectomy for recurrent giant cell tumor in the thoracic spine Case report MORIO MATSUMOTO, M.D., 1 KEN ISHII, M.D., 2 HIRONARI TAKAISHI, M.D., 2 MASAYA NAKAMURA, M.D., 2 HIDEO MORIOKA, M.D., 2 KAZUHIRO CHIBA, M.D., 2 TAKESHI TAKAHATA, M.D., 3 AND YOSHIAKI TOYAMA, M.D. 2 Departments of 1 Musculoskeletal Reconstruction and Regeneration Surgery and 2 Orthopaedic Surgery, School of Medicine, Keio University, Tokyo; and 3 Department of Orthopaedic Surgery, Isehara Kyodo Hospital, Kanagawa, Japan The authors report the case of a 47-year-old woman who harbored a giant cell tumor at the T-5 level. She had undergone curettage of the tumor via a combined anterior and posterior approach at a regional hospital and was later referred to the authors institution for treatment after the tumor recurred. On examination she exhibited progressive paraparesis and was nonambulatory due to cord compression caused by the tumor, which had invaded the spinal canal and extended to the right paravertebral muscles and right thoracic cavity. A spondylectomy was performed through a single posterior approach. The tumor, together with a portion of the dura mater, pleura, and muscles, was resected en bloc from T-4 to T-6. After resection, spinal reconstruction was performed by placement of an anterior titanium mesh cage as well as posterior pedicle screw and rod instrumentation. The patient s postoperative course was uneventful, and she exhibited substantial neurological recovery and became ambulatory. Two and a half years after surgery, the patient was tumor free. En bloc resection of a recurrent giant cell tumor was successfully achieved through a single posterior approach. This surgical technique can be an effective option for this pathological condition, which is difficult to manage using other conventional treatment options including repeated curettage and radiotherapy. KEY WORDS giant cell tumor thoracic spine tumor recurrence total spondylectomy A giant cell tumor is a locally aggressive tumor that arises predominantly in the sacrum and pelvis; its appearance in the upper spine is relatively rare. 15 The most common surgical option for a spinal giant cell tumor is curettage; however, incomplete removal of the tumor usually results in local recurrence and occasionally metastasis to the lung. 2,7 In their article, Sanjay et al. 9 reported that 10 of their 24 patients who harbored spinal giant cell tumors experienced local recurrence. Hart et al. 5 reported a recurrence rate of 28% in 36 cases of spinal giant cell tumors. This rate was considered lower than that for these tumors located in the extremities. Aggressive curettage of spinal giant cell tumor poses a great challenge to spine surgeons because of the risk of massive intraoperative bleeding due to hypervascularity of the tumor as well as the anatomical limitations provided by the intra- and paraspinal structures including the spinal cord, great vessels, and viscera. Although some authors have recommended aggressive curettage for spinal giant Abbreviations used in this paper: CSF = cerebrospinal fluid; MR = magnetic resonance; VB = vertebral body. cell tumors, 12 other authors have suggested undertaking en bloc resection or en bloc spondylectomy to reduce the amount of blood loss and avoid tumor contamination. 1,3,4,12 The optimal surgical procedure for recurrent spinal giant cell tumors has not yet been established. Repeated curettage may be inadequate to achieve complete resection of the tumor 14 and attain remission of the disease given the extent of the adhesions surrounding the recurrent lesion. Therefore, en bloc resection may be preferrable to curettage for recurrent giant cell tumors, although the latter remains the more common procedure for primary giant cell tumors. 3 In this report, we present the case of a patient with a recurrent giant cell tumor in the thoracic spine who was successfully treated using an extensive total en bloc spondylectomy through a single posterior approach. Case Report History. This 47-year-old woman presented to a regional hospital with a 6-month history of back pain, lower-extremity weakness, and difficulty in walking. At that time MR imaging studies revealed a T-5 spinal tumor (Fig. 1). 600 J. Neurosurg: Spine / Volume 6 / June, 2007

Giant cell tumor in the thoracic spine FIG. 1. Axial T 2 -weighted MR image obtained before the first surgery, demonstrating the spinal tumor involvement at T-5 compressing the spinal cord.

A laminectomy as well as posterior instrumentation with a titanium Luque rod was performed first, followed by curettage of the tumor and spinal reconstruction via an anterior extrapleural approach in

2 Giant cell tumor in the thoracic spine FIG. 1. Axial T 2 -weighted MR image obtained before the first surgery, demonstrating the spinal tumor involvement at T-5 compressing the spinal cord. She was admitted to the hospital and underwent an open biopsy procedure. A histopathological examination of the biopsy specimen revealed a giant cell tumor, and the patient underwent staged surgery. A laminectomy as well as posterior instrumentation with a titanium Luque rod was performed first, followed by curettage of the tumor and spinal reconstruction via an anterior extrapleural approach in which a titanium mesh cage packed with autologous minced rib was placed (Fig. 2). Her neurological condition improved after the surgery, and she regained ambulation. At 14 months after the initial surgery, the patient again developed lower-extremity weakness. She presented to the regional hospital, and tumor recurrence was noted. Because of rapid neurological deterioration, posterior decompression surgery was undertaken, but the surgery was interrupted because there was severe bleeding from the tumor. One month after the second surgery, she was referred to our hospital. Presentation and Examination. On admission the patient was unable to walk because of lower-extremity spastic paralysis, and she exhibited sensory disturbance below the level of T-7 and mild bladder dysfunction. Sagittal MR images revealed involvement of the tumor at the T4 6 vertebrae, and axial MR images demonstrated that the recurrent tumor extended into the right thoracic cavity, posterior paravertebral muscles, and had invaded the spinal canal causing severe spinal cord compression (Fig. 3). Angiography showed marked hypervascularity of the tumor as it was fed by the bilateral fifth through seventh intercostal arteries. Operation. After embolization of the feeding arteries, surgery was performed. A skin incision was made along the FIG. 2. Lateral radiograph obtained after the first surgery, showing the placement of the titanium cage from T-4 to T-6 as well as Luque instrumentation. previous surgical scar on the patient s back. Although the back muscles on the left side were separated from the spine, those on the right were attached to the tumor. The previously placed Luque rod was cut and removed from the spine, and titanium screws were inserted into the T1 3 and T7 9 pedicles. The bases of the left fourth through sixth ribs were exposed and separated from the spine. On the right side, the ribs were cut 8 to 10 cm away from the midline to avoid violating the tumor mass. In accordance with the technique described by Tomita et al., 14 blunt dissection was undertaken to separate the pleura manually from the lateral wall of the spine on the left side. On the right side, however, the parietal pleura was incised longitudinally along the cut edges of the ribs and was left attached to the tumor, and a plane between the parietal and visceral pleural layers was separated (Fig. 4A). Anterior to the spine, the parietal pleura was incised again under thoracoscopic visualization to avoid injury to the great vessels. A thoracoscope was placed close to the right anterior corner of the T4 6 VBs so that we could incise the parietal pleura in a well-illuminated and magnified surgical field and thereby safely reach the anterior cortex of the VBs. The tumor occupying the spinal canal was separated from the spinal cord by creating a plane between the outer and inner layers of the dura mater, leaving the outer layer of the dura attached to the tumor, to achieve resection of the J. Neurosurg: Spine / Volume 6 / June,

3 M. Matsumoto et al. FIG. 3. Sagittal Gd-enhanced T 1 -weighted (left) and axial T 2 -weighted (right) MR images showing the recurrent tumor involving the T4 6 levels and extending into the right paravertebral muscles, epidural space, and right thoracic cavity. The axial image was obtained at the T-5 level. epidural portion of the tumor s margins. A longitudinal incision was made on the left side of the posterior dura mater with the aid of a No. 15 rounded-blade knife, where the dura mater was free of adhesions and tumor. The outer layer of the dura mater was then dissected from the inner layer first using two pairs of microforceps, and then using a microdissector on the right side (Fig. 4B). At the points at which the right T4 6 nerve roots emerged from the dural tube, the roots were meticulously coagulated as was the outer layer of the dura mater and surrounding scar tissue to avoid contamination by tumor cells, and then they were ligated and severed. The interlayer dissection was continued up to the anterior portion of the spinal cord, and then the outer layer of the dura mater was incised longitudinally, again using electrocautery to reach the anterior epidural space, which was free of tumor. The left thoracic nerve roots were also ligated and severed outside the spinal canal. The anterior part of the dural sac was separated from the dorsal cortex of the VBs by using a spatula. When the separation was hindered by the presence of scar tissue, dissection between the scar tissue and the dura was performed using low-power electrocautery. Finally, complete separation of the anterior part of the dura and posterior cortex of the VBs was accomplished. No CSF leakage was observed at this point. Two threadwire saws were then passed around the spinal column at the levels of T3 4 and T After placement of the temporary rod, the thoracic spine was divided completely using the threadwire saws under fluoroscopic guidance. After rostrally and caudally dividing the spinal segments invaded by the tumor, the segments as well as the tumor were removed en bloc (Figs. 4C and 5). Permanent rods were placed bilaterally and secured with the pedicle screws. A titanium mesh cage packed with pasteurized local bone was placed at the level of the T3 7 vertebrae, and compression force was applied to the pedicle screws. The total surgical time was 14 hours and 30 minutes, and the total amount of blood loss was 6900 g. Postoperative Course. The patient s postoperative course was uneventful, except for the development of an asymptomatic CSF collection in the dead space after tumor excision. Given that she had no symptoms related to the CSF collection, she was carefully observed and did not undergo any interventional procedure, such as subarachnoid drainage. A good neurological recovery was obtained. At the follow-up examination conducted 2 years after the surgery, the patient was ambulatory without any assistance (Figs. 6 and 7). There was no evidence of tumor recurrence at the final follow-up examination performed 2.5 years after the surgery. Histopathological Examination. A histopathological investigation revealed that the surgical margin was free of tumor, except at one site facing the epidural space. The specimen showed abundant proliferating multinuclear oval giant cells (Fig. 8), and the tumor was diagnosed as a typical Grade II giant cell tumor. No evidence of malignant transformation was noted. 602 J. Neurosurg: Spine / Volume 6 / June, 2007

Giant cell tumor in the thoracic spine FIG. 5. Radiograph of the surgical specimen including the T4 6 spine, the titanium mesh cage placed in the previous surgery, ribs, and surrounding muscles. FIG. 4.

C = caudal; L = left; R = right. Discussion Few reports have been published on the treatment for recurrent giant cell tumors.

4 Giant cell tumor in the thoracic spine FIG. 5. Radiograph of the surgical specimen including the T4 6 spine, the titanium mesh cage placed in the previous surgery, ribs, and surrounding muscles. FIG. 4. Intraoperative photographs showing the separation of the tumor with the attached parietal pleura (A), the separated outer layer of the dura mater (B, arrows), and total removal of the tumor (C). C = caudal; L = left; R = right. Discussion Few reports have been published on the treatment for recurrent giant cell tumors. Shikata, 10 Ross, 8 and Fidler 3 and their colleagues reported successful results after using a combined anterior posterior approach for recurrent giant cell tumors in the thoracolumbar spine. Radiotherapy for recurrent spinal giant cell tumors is sometimes recommended because surgical removal can be difficult, 11 although its effectiveness in achieving tumor remission is uncertain and the exposure to radiation is also associated with adverse effects such as malignant transformation of the tumor. Tomita and colleagues 14 reported total en bloc spondylectomy for primary malignant spinal tumors. They achieved resection of the tumor margins through a single posterior approach except at the pedicle portion where the anterior and posterior parts of the spine are divided. For our patient s treatment, we adopted Tomita s technique with some modifications to deal with the expansive extension of the recurrent tumor. There were several technical difficulties that needed to be overcome. First, it was assumed that the resection of the epidural portion of the tumor would be difficult because of the severe adhesions of the tumor to the surrounding neural tissues after the previous two surgeries. Krepler et al. 6 reported a case of osteosarcoma of the thoracic spine that was treated by a wide resection of the T4 7 elements combined with resection of the dorsal part of the dura supplemented by Lyodura. Resection of all layers of the dura mater may be more radical; however, there exists the risk of contamination of the subarachnoid space with tumor cells or infectious meningitis. Our technique, in which the outer and inner layers of the dura are separated, precludes such risks. Resection of the tumor margins with the outer layer of the dura attached is possible, despite the presence of adhesions, whereas the inner layer of the dura and the arachnoid membrane are still preserved to avoid tumor cell contamination. Second, blunt dissection between the pleura and the tumor, which can be performed without much difficulty in J. Neurosurg: Spine / Volume 6 / June,

M. Matsumoto et al. FIG. 8. Photomicrograph showing a giant cell tumor and surrounding scar tissue (asterisk) and outer layer of the dura mater (arrowheads). H & E, original magnification 40. FIG. 6.

previously untreated patients, was problematic in our patient because of the presence of scar adhesions after repeated surgeries.

Circumferential dissection can be achieved between the VB and the surrounding pleura and mediastinal organs.

5 M. Matsumoto et al. FIG. 8. Photomicrograph showing a giant cell tumor and surrounding scar tissue (asterisk) and outer layer of the dura mater (arrowheads). H & E, original magnification 40. FIG. 6. Anteroposterior (left) and lateral (right) radiographs obtained 2 years after surgery. FIG. 7. Photograph showing the patient walking without aid or a cane 2 years after surgery. previously untreated patients, was problematic in our patient because of the presence of scar adhesions after repeated surgeries. In initial operations, surgeons can bluntly dissect the plane between the VB and pleura to reach the anterior aspect of the VB. Circumferential dissection can be achieved between the VB and the surrounding pleura and mediastinal organs. In the present case, on the right side of the tumor we divided the plane between the parietal and visceral pleura in the thoracic cavity, whereas on the left, we divided the ordinal plane between the VB and the parietal pleura. Thus, the dissection planes were different between the right and left sides, and we had to dissect the parietal pleura again at the front of the VB on the right side. To avoid injury to the vital organs in the mediastinum, including the azygos vein, we divided the ventral part of the parietal pleura under video-assisted thoracoscopic visualization and achieved circumferential separation of the VB from the surrounding tissues. A combined anterior posterior approach may be more commonly used than our single posterior approach. However, the wide tumor extension into the right paravertebral muscles, epidural space, and right thoracic cavity, as well as the adhesions to the right thoracic cavity due to the previous surgery in our patient, could have made the combined approach rather complicated, resulting in intralesional, suboptimal tumor resection. In conclusion, we have described a case of recurrent giant cell tumor which was successfully treated by total en bloc spondylectomy using a modified Tomita technique. This single posterior approach is useful for treatment of recurrent giant cell tumor, as it allows marginal resection of the tumor, which is mandatory to prevent further recurrence. References 1. Abe E, Sato K, Tazawa H, Murai H, Okada K, Shimada Y, et al: Total spondylectomy for primary tumor of the thoracolumbar spine. Spinal Cord 38: , Chen LH, Niu CC, Lai PL, Fu TS, Chen WJ: Recurrent giant cell 604 J. Neurosurg: Spine / Volume 6 / June, 2007

6 Giant cell tumor in the thoracic spine tumor of the thoracic spine with bilateral pulmonary metastases. J Formos Med Assoc 103: , Fidler MW: Surgical treatment of giant cell tumours of the thoracic and lumbar spine: report of nine patients. Eur Spine J 10:69 77, Gille O, Soderlund C, Berge J, Sacko O, Vital JM: Triple total cervical vertebrectomy for a giant cell tumor: case report. Spine 30: E272 E275, 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 22: , Krepler P, Windhager R, Toma CD, Kitz K, Kotz R: Dura resection in combination with en bloc spondylectomy for primary malignant tumors of the spine. Spine 28:E334 E338, Ozaki T, Liljenqvist U, Halm H, Hillmann A, Gosheger G, Winkelmann W: Giant cell tumor of the spine. Clin Orthop Relat Res 401: , Ross AE, Bojescul JA, Kuklo TR: Giant cell tumor: a case report of recurrence during pregnancy. Spine 30:E332 E335, Sanjay BK, Sim FH, Unni KK, McLeod RA, Klassen RA: Giantcell tumours of the spine. J Bone Joint Surg Br 75: , Shikata J, Yamamuro T, Shimizu K, Shimizu K, Kotoura Y: Surgical treatment of giant-cell tumors of the spine. Clin Orthop Relat Res 278:29 36, Shoda N, Nakajima S, Seichi A, Kan A, Iwasaki M, Kitagawa T, et al: Computer-assisted anterior spinal surgery for a case of recurrent giant cell tumor. J Orthop Sci 7: , Solini A, Gargiulo G: Giant cell tumor of the spine. Surgical findings. Chir Organi Mov 83:35 42, Tomita K, Kawahara N: The threadwire saw: a new device for cutting bone. J Bone Joint Surg Am 78: , 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 22: , Turcotte RE, Sim FH, Unni KK: Giant cell tumor of the sacrum. Clin Orthop Relat Res 291: , 1993 Manuscript submitted August 18, Accepted March 5, Address reprint requests to: Morio Matsumoto, M.D., Department of Orthopaedic Surgery, School of Medicine, Keio University, 35 Shinanomachi, Shinjuku-ku, Tokyo, , Japan. morio@sc.itc.keio.ac.jp. J. Neurosurg: Spine / Volume 6 / June,

En bloc spondylectomy has been developed and. Late instrumentation failure after total en bloc spondylectomy. Clinical article

En bloc spondylectomy has been developed and. Late instrumentation failure after total en bloc spondylectomy. Clinical article J Neurosurg Spine 15:320 327, 2011 Late instrumentation failure after total en bloc spondylectomy Clinical article Morio Matsumoto, M.D., 1 Kota Watanabe, M.D., 2 Takashi Tsuji, M.D., 1 Ken Ishii, M.D.,