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Multilevel Pedicular Osteotomies for en-bloc Resection of a Primary Ewing Sarcoma


of the Subaxial Cervical Spine with Pedicle Screw Reconstruction

Julien Francisco Zaldivar-Jolissaint, Lukas Bobinski, MD, John Michael Duff, MD


FACS

PII: S1878-8750(19)32124-2
DOI: https://doi.org/10.1016/j.wneu.2019.07.208
Reference: WNEU 13008

To appear in: World Neurosurgery

Received Date: 9 July 2019

Accepted Date: 28 July 2019

Please cite this article as: Zaldivar-Jolissaint JF, Bobinski L, Duff JM, Multilevel Pedicular Osteotomies
for en-bloc Resection of a Primary Ewing Sarcoma of the Subaxial Cervical Spine with Pedicle Screw
Reconstruction, World Neurosurgery (2019), doi: https://doi.org/10.1016/j.wneu.2019.07.208.

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© 2019 Published by Elsevier Inc.


1 World Neurosurgery
2
3 Technical Note
4
5 Multilevel Pedicular Osteotomies for en-bloc Resection of a Primary
6 Ewing Sarcoma of the Subaxial Cervical Spine with Pedicle Screw
7 Reconstruction.
8
9
10 Julien Francisco Zaldivar-Jolissaint1; Lukas Bobinski, MD2; and John Michael
11 Duff, MD FACS3
12
13
14
1
15 Service de Neurochirurgie, Centre Hospitalier Universitaire de Grenoble Alpes
16 (CHUGA), La Tronche, France
2
17 Department of Orthopedic Surgery, Spine Unit, University Hospital of Umea, Umea,
18 Sweden
3
19 Neurosurgeon, Clinique de Genolier, Genolier, Switzerland
20
21
22
23 Information:
24 Running Title: Cervical Ewing sarcoma excision and spine stabilization
25 Keywords: Ewing Sarcoma; Cervical Spine; Cervical Pedicle Screw Stabilization; En-bloc
26 Excision, Multilevel Cervical Pedicular Osteotomies
27 Words in abstract: 170
28 Words in article: 1140
29 Image format: jpeg / 150 dpi for B&W and color. Images were edited using Adobe Illustrator.
30 Number of References: 23
31
32 Corresponding author:
33 Service de Neurochirurgie, Centre Hospitalier Universitaire de Grenoble Alpes (CHUGA)
34 Avenue Maquis du Grésivaudan, 38700 La Tronche, France
35 julienzaldivar@gmail.com
36
37 Contributorship Statement:
38 All authors were involved in patient care.
39 Julien Francisco Zaldivar-Jolissaint co-drafted the manuscript and built the figures.
40 Lukas Bobinski co-drafted the manuscript, edited the figures and provided key bibliographic
41 input to the writing.
42 John Michael Duff co-drafted the manuscript, edited the figures and provided final manuscript
43 revision.
44
45 Consent:
46 The authors declare that there is no information in the manuscript that could break patient
47 anonymity or allow identification.
48
49 Disclosures:
50 The authors declare no financial conflict of interest.
51

1
1 Abstract

2 Background

3 Primary Ewing Sarcoma of the mobile spine is a rare disease. Its

4 management requires careful surgical planning as radical, margin-free

5 excision is directly correlated to prognosis. Extensive bone removal in the

6 cervical spine can lead to instability and cause postoperative, iatrogenic

7 cervical deformity. Spinal instrumentation thus plays an important role in

8 restoring post-resection spinal stability and improving quality of life.

9 Methods

10 The authors present a novel technique that allowed successful removal of a

11 large Ewing Sarcoma of the subaxial cervical spine, infiltrating and traversing

12 the posterior bone elements, and extending into the paraspinal muscles.

13 With this technique, radical en-bloc resection of posteriorly located cervical

14 tumors via multilevel pediculotomies is described, with terminal vertebrae

15 pedicle screw reconstruction.

16 Results

17 Terminal vertebrae cervical pedicle screw reconstruction allowed wide

18 surgical excision with satisfactory oncological and mechanical results.

19 Conclusions

20 The described technique enables one stage total tumor resection and

21 stabilization. It may be a viable alternative in selected cases for radical en

22 bloc resection of posteriorly located epidural malignant lesions of the cervical

23 spine.

2
1 Introduction

2 Primary Vertebral Ewing Sarcoma (PVES) is a rare disease1. It accounts for

3 only 10% of histologically confirmed Ewing sarcomas and occurs with a peak

4 incidence in the second decade of life1. It preferentially involves the sacral

5 spine (53%), followed by the lumbar (25%), thoracic (11%) and only rarely the

6 cervical spine (3%), with the remaining involving more than one spinal region

7 at presentation (8%)1. The most common presenting symptom in malignant

8 primary spine tumors is local pain which arises classically during rest and at

9 night, typically independent of mechanical loading2. Other symptoms may

10 include progressive neurological deficit, radicular pain and/or myelopathy due

11 to compression of the neural elements.

12 For primary bone tumors, the surgical challenge is the feasibility of en bloc

13 tumor resection, as tumor free margins are directly correlated with local

14 recurrence and tumor-related death3–7. Overall predictive median survival is

15 highest in patients who undergo tumor-free margin excision 8,9. Another series

16 demonstrates that sub-optimal intra-lesional resection combined with adjuvant

17 chemotherapy and radiotherapy has poorer outcomes when compared to

18 radiotherapy and chemotherapy alone 4, further underlining the importance of

19 avoiding tumor margin violation. Radical en bloc surgical tumor excision with

20 tumor-free margins is desirable when technically feasible for primary bone

21 tumors in the spine. However, this strategy may induce spinal instability, as in

22 our case. Indeed, iatrogenic spinal deformity following surgical excision is one

23 of the most common complications after excisional surgery for Ewing sarcoma

24 in the mobile cervical and lumbar spine9.

3
1 The authors present a case of a cervical extradural, intraspinal and paraspinal

2 Ewing sarcoma, which was resected using multilevel transpedicular

3 osteotomies to “disconnect” the posterior elements from their respective

4 vertebral bodies and allow en bloc resection with subsequent cervical pedicle

5 screw stabilization. To our knowledge, this is the first English language report

6 of this technique for managing primary malignant tumor of the posterior

7 elements of the subaxial cervical spine.

9 Case Description

10 A 17-year old male presented to the emergency department of the University

11 Hospital of Lausanne with a three-week-history of rapidly progressive neck

12 pain irradiating to both arms, with upper limb paresthesias. Neurologic

13 examination confirmed a mild cervical myelopathy with a modified Japanese

14 Orthopedic Association score (mJOA) of 15. Magnetic Resonance Imaging

15 (MRI) and Computed-Tomography (CT) revealed a contrast enhancing,

16 extradural, paraspinal mass causing extensive spinal cord compression,

17 traversing the bony posterior elements into the paraspinal muscles, and

18 extending from C5 to C7 (Figure 1A-F). Contrast enhanced CT of the chest

19 and abdomen were normal. Routine blood laboratory values were normal.

20 Because of recent onset of progressive myelopathy, a single-stage posterior

21 en-bloc resection combined with cervical pedicle screw (CPS) reconstruction

22 was planned without prior biopsy.

23 The patient was positioned in the prone position on the Jackson table with the

24 head fixed in the Mayfield holder and the cervical spine in a neutral position. A

4
1 standard midline posterior skin incision from the occiput to T3 was performed.

2 The specific surgical steps are as follows.

3 -Skin and subcutaneous flaps were raised on both sides prior to fascial

4 opening. The fascia was incised approximately 1 cm from the midline

5 bilaterally. The trans-muscular dissection was carried down to the lateral

6 border of the facets to avoid entering into the tumor-infiltrated muscles

7 (Figure 2).

8 -Following exposure of the lateral aspects of the C3/4 and C7/T1 facet joints

9 bilaterally, C4 and T1 pedicle screws were placed bilaterally using image

10 guidance (O-Arm®).

11 -The posterior elements were disconnected by performing cervical “Ponte like”

12 osteotomies10 at the terminal segments below the C4, and above the T1

13 pedicles bilaterally.

14 -At the intermediate segments, the posterolateral portions of the facets were

15 drilled away. Lateral to medial transpedicular osteotomies were performed

16 bilaterally across the proximal part of the C5, C6 and C7 pedicles with a high-

17 speed drill (Figure 2 and Figure 3)

18 -Final bone disconnection was achieved by dividing the medial cortical walls

19 of the pedicles with a 1mm Kerrison punch.

20 -Ligamentous attachments laterally and at the superior and inferior ends were

21 divided with a small Kerrison and a microscissors allowing progressive

22 elevation and eventual en bloc removal of the tumor specimen (Figure 2 C).

23 The tumor capsule was in direct contact with the posterior dura but could be

24 lifted off it without difficulty (Figure 2 B-D). There was no macroscopic

25 evidence of adherence or invasion. No attempt was made to resect the

5
1 adjacent dura which looked normal. The ventral tumor surface was not

2 macroscopically violated at any point.

3 -Spinal stabilization was then performed using bilateral lordotic rods

4 connecting the pedicle screws placed at C4 and T1 bilaterally. No

5 intermediate fixation screws were possible at the C5, C6 and C7 pedicles, as

6 they had been resected as part of the specimen.

7 -Allograft and demineralized bone matrix were used as a bone fusion

8 substrate.

9 Pathological examination revealed Ewing’s Sarcoma with macroscopic tumor

10 free margins. Histologically, there was a possible marginal violation at a single

11 pedicular level, though this was uncertain.

12 The patient made an excellent recovery with a normal neurological

13 examination postoperatively. He left the hospital 8 days after the surgery and

14 subsequently underwent radiotherapy and chemotherapy. At 2 year follow-up,

15 the patient was fully ambulant, with a normal neurological clinical examination

16 and only mild residual neck pain with no signs of myelopathy (mJOA 18).

17 There were no signs of instability but posterior paraspinal muscle atrophy was

18 clearly visible. Contrast enhanced MRI showed no local recurrence. CT scan

19 and standard x-rays with flexion/extension showed good spinal alignment and

20 osseous fusion without any signs of hardware failure (Figure 4).

21

22 Discussion:

23 Surgical treatment of primary malignant spine tumors is very challenging. In

24 such cases, the prognosis is related to how radical the surgical resection is,

25 with the goal of achieving en bloc resection with tumor-free margins3–7,11.

6
1 Infiltration of surrounding structures (dura, spinal cord, nerve roots, vessels)

2 makes complete resection technically difficult and often impossible without

3 unacceptable morbidity12. Moreover, the wide excision of the tumor with

4 surrounding structures (paraspinal muscles, lamina, lateral masses, ligaments

5 and spinal joints) can cause iatrogenic post-operative pain, instability and

6 spinal deformity13–16.

7 Posterior pedicle screw fixation after tumor resection has been shown to be a

8 valuable tool to prevent instability in metastatic disease17,18. The use of CPS

9 is advantageous because of high resistance to pullout forces and is

10 independent from the amount of bone in the lateral masses19–23, thus

11 providing immediate postoperative stability and the possibility of long-term

12 bone fusion. In our case, using CPS instrumentation at C4 and T1 levels

13 facilitated reconstruction following complete en-bloc resection over three

14 segments (C5-C7). No intermediate screws were placed as the facets and

15 pedicles at these levels had been partially resected as part of the specimen.

16 No instability or hardware failure was seen on follow-up.

17 The combination of trans-pedicular osteotomies and CPS reconstruction in

18 the cervical spine may be a useful option in cases where en bloc resection of

19 posteriorly located tumors and stabilization is required.

20

7
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1 transpedicular screw fixation in the cervical spine. Spine.
2 1994;19(22):2529-2539.

10
1 Figure Legends
2
3 Figure 1
4 A. T1-weighted gadolinum-enhanced sagittal view showing a posteriorly lying
5 extradural mass with contrast enhancement extending from C5 to C7, and
6 thecal sac and spinal cord compression.
7 B. T1-weighted gadolinum-enhanced axial view at the C6 level showing tumor
8 infiltrating the posterior elements and into the para-vertebral musculature.
9 C. T2-weighted sagittal view showing the tumor mass from C5 to C7.
10 D. T2-weighted axial view at the C6 level.
11
12 Figure 2
13 A. Schematic diagram of the soft tissue resection and pedicle osteotomies. B.
14 Traction of the tumor specimen (arrowhead) allows microsurgical dissection
15 with dural visualization (*)
16 C. Excised tumor
17 D. Post-excisional decompression. The dura appears macroscopically normal
18 (*)
19
20 Figure 3
21 Immediate post-operative CT.
22 A. Sagittal Midline CT showing level of axial CT cuts.
23 B. Sagittal trans-articular CT showing the extent of the posterior element bony
24 removal, with bilateral pedicular osteotomies.
25 C. D and E. Axial CT showing the complete pedicular osteotomies at the C5,
26 C6 and C7 vertebrae respectively (*). Bone graft material is also visible.
27
28 Figure 4
29 A. 2-year Follow-up axial (C6) and coronal CT showing extensive fusion
30 (arrowheads)
31 B. 2-year Follow-up Antero-posterior and lateral standing X-rays showing
32 good spinal alignment, hardware position and bone fusion.

11
A

B C

*
A B

C D
A
A C B
D
E

C D E

* * * *
* *
A

B
1 World Neurosurgery
2
3 Abbreviations
4
5 Multilevel Pedicular Osteotomies for en-bloc Resection of a Primary
6 Ewing Sarcoma of the Subaxial Cervical Spine with Pedicle Screw
7 Reconstruction.
8
9 Abbreviation Signification
10 CPS Cervical Pedicle Screw
11 CT Computed-Tomography
12 mJOA modified Japanese Orthopedic Association score
13 MRI Magnetic Resonance Imaging
14 PVES Primary Vertebral Ewing Sarcoma

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