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Reliable Intraoperative Repair Nuances of Cerebrospinal Fluid Leak in Anterior Cervical Spine Surgery and Review of the Literature
Accepted Manuscript Reliable Intraoperative Repair Nuances of CSF Leak in Anterior Cervical Spine Surgery and Review of The Literature Bartley D. Mitchell, MD PhD, Terence Verla, MD, MPH, Duemani Reddy, MD PhD, Lona Winnegan, RN, Ibrahim Omeis, MD PII:
S1878-8750(16)00093-0
DOI:
10.1016/j.wneu.2016.01.014
Reference:
WNEU 3611
To appear in:
World Neurosurgery
Received Date: 23 November 2015 Revised Date:
11 January 2016
Accepted Date: 11 January 2016
Please cite this article as: Mitchell BD, Verla T, Reddy D, Winnegan L, Omeis I, Reliable Intraoperative Repair Nuances of CSF Leak in Anterior Cervical Spine Surgery and Review of The Literature, World Neurosurgery (2016), doi: 10.1016/j.wneu.2016.01.014. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Reliable Intraoperative Repair Nuances of CSF Leak in Anterior Cervical Spine Surgery and Review of The Literature
Winnegan RN1, Ibrahim Omeis MD1
Department of Neurosurgery, Baylor College of Medicine, 7200 Cambridge St,
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Bartley D. Mitchell MD PhD1, Terence Verla, MD, MPH1, Duemani Reddy MD PhD1, Lona
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Suite 9A, Houston, TX 77030
Corresponding Author:
Ibrahim Omeis MD
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Department of Neurosurgery Baylor College of Medicine
7200 Cambridge Street, suite 9A Houston, TX 77030
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Email: [email protected]
Key words: Anterior cervical spine; CSF leak; direct repair. Abbreviations: ACDF=Anterior cervical discectomy and fusion; AI=anterior instrumentation; CSF=Cerebrospinal fluid; NF=Neurofibromatosis; OPLL=Ossification of Posterior Longitudinal Ligament; PIF=posterior instrumented fusion; HLOS=Hospital length of stay; F/U=Follow up duration; WP=Wound peritoneal; LP=Lumbar peritoneal
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INTRODUCTION Cerebrospinal fluid leak during anterior cervical spine surgery is a known potential complication, and must be treated in order to prevent additional complications, including
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dysphagia, airway compromise, headaches, failed fusion, poor wound healing, or even
meningitis1-5. Although the overall incidence of CSF leak during anterior cervical spine surgery has been reported to occur in 0.2 to 1% of cases of Anterior Cervical Discectomy and Fusion
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(ACDF) or degenerative cervical spine operations6-8, there are conditions that may allow for higher rates of CSF leak during anterior cervical spine surgery, including ossification of the
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posterior longitudinal ligament (OPLL), dural ectasia in Neurofibromatosis type 1 or Marfan’s disease, intradural disk herniation, and compressive osteophytes6,7,9-13. In these cases, the dura is often tenuous at best or directly attached to the posterior longitudinal ligament, and frequently does not lend itself to primary closure, given that large portions of the dura may be missing
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altogether. There are multiple methods for treating cerebrospinal fluid leaks encountered during anterior cervical spine surgery, including primary dural closure (in cases where the dural edges are able to be sutured), fibrin glue, gel-foam, fat and fascial grafts, muscle grafts, blood patch,
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lumbar drainage, and post-operative bed positioning, and most of these methods are described in combination2,8,11,13-20 These methods have been found to be successful in many cases,
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particularly with otherwise uncomplicated anterior cervical spine surgery for ACDF, OPLL, or degenerative cervical spine. However, in cases with particularly expansive durotomies or with friable dural tissue, an alternative solution is often necessary. Herein, we present a series of 8 patients who underwent anterior cervical spinal surgery
and experienced an intra-operative CSF leak with either a large gap between dural edges or with friable or frail dural edges that could not be primarily repaired. We employed a unique
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combination therapy in the management of the CSF leak while avoiding associated complications and post-operative symptoms. Likewise, extensive review of the literature was performed to identify the different methods of treating anterior CSF leak and to compare the risk
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of failure with our method.
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METHODS In this retrospective case series, patients were included if they had: 1) undergone an anterior cervical spine surgery for one of the following conditions considered high-risk for
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durotomy and CSF leak: OPLL, Neurofibromatosis with dural ectasia, intradural disc
herniations, and compressive osteophytes, and 2) experienced a CSF leak that was recognized and treated intra-operatively. Eight patients met these criteria from this single-institution series.
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Charts were reviewed in these cases, and the methods used for repair of the CSF leak/durotomy were reviewed, along with patient outcomes and any complications that were potentially related
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to the surgical procedure. Improvement in functional outcomes was evaluated by comparing the pre and post-operative Visual Analogue Scale (VAS) and the Neck Disability Index (NDI). In each case, the same technique was used when a large CSF leak was encountered without the
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possibility of primary repair:
Intraoperative Nuances employed for dural repair: 1) Intentional drainage of CSF was performed using a cottonoid and a very low pressure
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suction to the point whereby no fluid was seen in the surgical field with the patient in a trendelenburg position, in order to keep the area around the dural defect as dry as
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possible.
2) Placement of a dural substitute, Durepair (Medtronic; Minneapolis, Minnesota), large enough to cover the entire exposed area of dura and if possible to tuck it under the adjacent bones.
3) Dry the exposed surface of the Durepair with a cottonoid and light suction.
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4) Apply DuraSeal (Covidien/Medtronic; Minneapolis, Minnesota) over the area of the dural defect with Durepair onlays, while taking care not to fill the entire expanse of the surgical cavity, given the possibility of expansion of the product, with possible dural and
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spinal cord compression.
5) Use a high-speed drill with a cutting burr to shave and shape the interbody implant on the side that will be facing towards the dura, such that it overlaps approximately the anterior
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half of the end-plate surface area.
6) Place the implant and secure the construct with an anterior titanium plating system into
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the vertebral body above and below the repair/implant site. Figure 1, part D shows the contoured implant secured with anterior instrumentation.
7) Place a tunneled percutaneous passive drain system connected to a drain bag. The catheter was placed in the surgical bed to prevent fluid accumulation/hematoma and not
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to divert CSF. The drain usually stays in place for 2 days.
All patients were allowed to sit up 3-4 hours post-operatively, and were encouraged to
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ambulate and mobilize on post-operative day 1. When the drains were removed, a nylon suture was placed at the exit site in all cases. Patients were then monitored for signs of intracranial
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hypotension, neck swelling, airway compromise, dysphagia, or meningitic symptoms, both during their hospitalization time and in the ambulatory follow-up.
Cases examples: Case 1
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80-year-old female presented with difficulty walking and weakness. Imaging studies revealed a significant C5-6 disk herniation with cord compression (Figure 1), and surgery was offered to her. Intra-operatively, there was a large defect in the dura at the C5-6 level that could
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not be repaired primarily with sutures only. The dura was opened intentionally to resect the intradural disc herniation. Given the disc was adherent to the ventral surface of the dura, it was resected with the herniated disc fragment leaving a wide opening in the dura. The dural opening
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was repaired as described above (Figure 2), with no subsequent leak. Post-operatively, there was no further drainage from the site noted. Her pain has improved as well as her functional
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mobility, and she was discharged home in good condition on post-operative day 3. Case 2
A 29 year-old man with a history of neurofibromatosis 1 and multiple neurofibromas presented with signs and symptoms of cervical myelopathy and pain radiating down his right
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upper extremity with weakness in his right upper extremity and difficulty walking for 2 weeks. CT and MRI of his cervical spine showed severe kyphotic deformity primarily at C5, with near complete subluxation and severe spinal cord compression, along with extensive dural ectasia
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(Figure 3). He underwent anterior decompression with C5 and 6 complete corpectomy with partial C4 and C7 corpectomies, with anterior and posterior instrumentation. During the anterior
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portion of the decompression, due to the dural ectasia, he had an almost non-existent dura with significant CSF leak, requiring repair as described in this paper. However, given the extent of the anterior decompression, we used an expandable cage, with a smaller diameter than we usually use to leave a space for the dural substitute/Sealant agent to expand and not cause any cord compression. Despite this, the patient did well and was able to ambulate freely post-
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operatively. He was discharged to an acute rehabilitation facility with no evidence of wound
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drainage or dehiscence. He had no evidence of dysphagia.
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RESULTS In this series, all 8 patients experienced an intra-operative CSF leak while undergoing anterior cervical spine surgery involving bony decompression with partial or complete
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corpectomy. All of the patient cases presented in this series were known to have risk factors for durotomy (NF1 with dural ectasia, OPLL, intradural disc herniation, or prominent posterior endplate osteophytes), and each had a durotomy that was not able to be closed primarily,
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necessitating an implementation of alternative strategy for prevention of CSF leak.
Table 1 shows the patient characteristics seen in this series. The patients were evenly divided by
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gender, and the age range was 30-80 years. The mean follow up duration was 31.8 months (Table 1). OPLL accounted for half the cases, the other half consisting of NF1, intradural disc herniation, and one severely ossified posterior disc osteophyte. The dural defect ranged from the size of the disc herniation of approximately 1cm to a large defect of approximately 3-4cm with
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paper-thin, floppy dural openings as in the case of the NF-1 patient. All of the patients underwent intraoperative treatment for their CSF leak as described above in the methods section. In this series using this repair protocol, there were no further CSF leaks, nor any associated
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complications typically associated with persistent cervical spine area CSF leaks. Despite draining CSF using low suction with the intradural compartment insulated with a cottonoid to achieve a
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dry surgical field, there were no complaints of headaches, nausea, vomiting or any other symptoms associated with intracranial hypotension post-operatively. All patients were monitored closely throughout their hospital stay, and all drains were removed by post-operative day 2. In the post-operative period, all patients received antibiotics until the drains were
removed. They also participated in physical therapy. The hospital length of stay ranged from 3 days (one level ACDF) to 7 days (three level Corpectomy with posterior instrumented fusion).
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By 6 months post-operatively, none of the patients had any documented complications suggesting persistent CSF leak, infections or wound breakdown. Overall, there was a significant improvement in the mean VAS scores (pre-op: 8.9 vs post-op: 1.9, p<0.0001) and NDI scores
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(pre-op: 29.4 vs post-op: 7.3, p<0.0001). Figure 4 shows the changes in the VAS and NDI scores
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following surgery utilizing the closure technique described in this study.
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DISCUSSION Anterior cervical spine surgery is a widely used surgical operation in patients with symptomatic cord compression or foraminal stenosis secondary to a variety of cervical
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pathologies including canal stenosis, myelopathy or instability21-23. Despite the success rate, there are some presenting pathologies such as OPLL, intradural disc herniation and compressive osteophyte whereby the dura mater becomes calcified and adheres to the posterior longitudinal
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ligament, increasing the risk for durotomy during anterior decompression. With the high
incidence of CSF leak with OPLL6,7,11,17,24, and with studies showing as high as 13.7% increased
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risk of CSF leak with OPLL during anterior cervical spine surgery13, we sought to describe a successful and reliable strategy for intra-operative repair of CSF leak to improve clinical outcome while reducing associated morbidity.
Each of the cases in this series was considered high risk for CSF leak because of pre-
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existing conditions such as OPLL, Neurofibromatosis with dural ectasia, intradural disk herniation, and large compressive osteophytes. In each case described, the durotomy was substantial, and illustrates the need for a reliable method of dural repair and prevention of further
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CSF leak, while still achieving the goals of the surgery, mainly decompressing the spinal cord and providing structural stability. There were no intra-operative or post-operative neurologic
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complications within 6 months in all the patients involved. No patient developed pseudarthrosis at last follow-up. Using the methods described here, we were able to achieve complete prevention of CSF leak in all cases, although the total number of cases is admittedly small. It is important to note that in each of these cases, the structural allograft used was shaped prior to implantation in order to allow more room between the structural graft and the patch materials, while still providing adequate spinal stability. This is crucial in cervical spine surgery given the
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anticipated expansion of the materials used in this algorithm, including gelfoam and fibrin glue. Without adding this extra space, it is possible for expansion of the dural repair products with compression of the spinal cord, causing severe progressing neurologic deficits, which has been
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previously reported in studies19,25.
In all of the cases described in this series, the type of Dura-Seal (Integra) used was
formulated for spinal use. This formulation differs from the cranial formulation in that although
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it does expand, it does not expand nearly to the same degree as the cranial type. Nevertheless, in performing this particular intra-operative CSF leak control method, we recommend allowing
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extra room for allograft expansion. Careful observation should be made in the immediate postoperative phase, with a high index of suspicion for graft-expansion in the event of rapid declining neurologic status. In addition, we intentionally drained CSF intraoperatively using a cottonoid and low pressure suction to create a relatively dry environment for the dural substitute
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to adhere to the dura once the sealant agent was applied. This avoids re-accumulation of CSF, which could potentially cause dehiscence of the patch. Several studies have reported different strategies for the management of intra-operative
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CSF leak during anterior cervical spine surgery with varying success rates. Intra-operative repair can be achieved using gelatin foam, fibrin glue, dural grafts, DuraSeal, muscle grafts or any
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combination of these2,8,13,15-20. Post-operatively, lumbar CSF drains and lumbar/wound-peritoneal shunts have been utilized as a preventive measures to reduce CSF leaks6,8,16,17,26-28. In a series by Hannallah et al13, 20 patients were identified with intra-operative CSF leak. Five patients with accessible dural tears underwent intraoperative repair with the use of anterior cervical drains kept on gravity-drainage and discontinued on post-operative day 2. Another series of 13 patients by Syre et al8 reported successful intraoperative repair of 8 CSF leaks using a combination of
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gelfoam, fibrin glue, muscle graft and Duragen, without need for additional treatment post-op. The remaining 5 patients required lumbar drains, re-exploration and shunts for persistent CSF leak. In addition to intraoperative repair, several studies have utilized placement of lumbar
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drains and lumbar/wound-peritoneal shunts to achieve satisfactory outcomes8,16,17,28-30. Table 2 shows a summary of clinical cases with intra-operative repair of CSF leaks with or without the placement of lumbar drains or shunts.8,13,16,17,20,28,31-33
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There may be no one single method for repairing large durotomies, and certainly the method described in our series may not work on all leaks. The method described here does not
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offer an entirely new concept either, rather it uses a particular combination of repair strategies combined with the use of intentional drainage of CSF to the point it becomes dry at the durotomy site followed by direct patching of the defect with a sealant. We believe extra drainage of CSF prior to the initiation of patching may give some time for the patch to adhere to the dura and
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prevent dehiscence by applying the sealant agent to cover it and provide some pressure. Then, utilizing a passive drain (drain to gravity, not suction) allowed for drainage of any excess fluid that may accumulate. This presumably may prevent complications caused by fluid compression
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of the trachea (respiratory distress), esophagus (dysphagia), or other structures, while preventing active siphoning of fluid, thus preventing the dural repair from truly sealing. This is similar to
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Hannallah et al13 in their approach to accessible dural tears with the use of gravity drainage, achieving satisfactory clinical outcomes. Furthermore, our protocol does not utilize lumbar drains and lumbar/wound-peritoneal shunts which can add its own set of complications11. In our study, there was a significant improvement in functional outcomes (VAS and NDI)
at 6 weeks following surgery and there was no post-operative complication in all eight patients. This illustrates the safety and reliability of our strategy at intraoperative management of CSF
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leaks in circumstances whereby the extent of the dural tear is not amendable to primary closure
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techniques.
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CONCLUSION Overall, this method was reliable and reproducible in 8 consecutive cases, with straightforward strategies and nuances that can be implemented readily during the encounter of
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an anterior cervical spine dural opening whether it was intentional or not. Although the
development of more methods for repair of large durotomies is certainly warranted, this method
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persistent CSF leaks from anterior spine surgery.
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may be a useful tool in any surgeon’s armamentarium to avoid morbidity associated with
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FIGURE LEGEND: Figure 1: (Case1) 80y/o F with significant C5-6 disk herniation with cord compression presenting with difficulty walking and weakness. She underwent C5-6 ACDF.
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(A) Sagittal, (B) Axial views of CT scan demonstrating the intradural calcified disc herniation. (C) Intra-operative X-Ray showing the position of the graft (arrow) shortened to allow expansion of the sealant agent.
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(D) Lateral XR-Ray with drawings showing the graft and materials used to seal the dural defect. (The graft (red) is reduced in size to allow for the sealant agent (Blue) and dural substitute
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(Brown) to expand without causing cord compression. (White) is space left between the graft and the sealant agent.
Figure 2: Intraoperative images showing
(A) Intradural calcified disc fragment (arrow: dural edge; asterisk: large intradural calcified
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fragment).
(B) Exposure of the spinal cord after resection of the disc fragment (C) Placement of a dry dural substitute, and (D) sealant agent.
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Figure 3: (Case 2) 29y/o M with history of NF-1, presents with severe kyphotic deformity, with near complete subluxation and severe spinal cord compression, along with extensive dural
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ectasia. He presented with cervical myelopathy and radiating pain down his upper extremity with weakness. He underwent cervical corpectomies followed by placement of expandable cage and C3 to T1 anterior instrumentation. Sagittal CT (A), MRI (B) showing the extensive pathology of neurofibroma, dural ectasia and subluxation. (C) post-op CT scan showing reconstruction of the anterior spine.
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Figure 4: Significant improvement in the pre- and post-operative VAS and NDI scores in patients undergoing intra-operative repair of CSF leak using the technique described in this
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study.
TABLE LEGEND
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Table 1: Characteristic summary of the 8 patients included in the series
Table 2: Clinical studies describing the management of intra-operative CSF leaks and their
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success rate
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Table 1: Characteristic summary of the 8 patients included in the series Gender
Presenting symptoms
Preoperative diagnosis
Level of surgery
1
80
F
29
M
Intradural disc herniation NF-1 with neurofibroma and dural ectasia
C5-6 ACDF
2
Weakness, walking difficulty Radiating neck pain
3
48
F
OPLL
4
70
M
Radiating neck pain, walking difficulty Walking Difficulty
5
54
M
6
47
Radiating neck pain Radiating neck pain, falls, walking difficulty
Compressive osteophyte OPLL and Compressive osteophyte
7
61
8
48
Duration of post-op wound drainage 2
Post-op complications (within 6mths)
HLOS (days)
F/U (mths)
Disposition
None
3
13
Home
None
2
None
7
42
Acute Rehab – Symptom resolution at last f/u
None
2
None
5
22
Home
None
2
4
51
Acute Rehab - Symptom resolution at last f/u Home
None
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OPLL
C4-7 corpectomies with C3-T1 anterior instrumentatio n C5 C with C4-6 AI followed by PIF C4-5 ACDF
Postop CSF leak None
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Age
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Case #
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C5-6 None 2 None 3 32 ACDF F C3-4 ACDF None 2 None 5 42 Patial C3, complete C4 C with C3-5 AI followed by PIF M Radiating OPLL and C4 C with None 2 None 5 28 neck pain, Compressive C3-5 AI with falls, osteophyte PIF F Numbness, OPLL C4-5 C None 2 None 6 25 weakness With C3-6 AI followed by PIF AI=anterior instrumentation; C=corpectomy; PIF=posterior instrumented fusion. OPLL=Ossification of Posterior Longitudinal Ligament; ACDF=Anterior cervical discectomy and fusion; NF=Neurofibromatosis; HLOS=Hospital Length of stay. F/U=Follow up duration
Home
Home
Home
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Table 2: Clinical Studies describing the management of intra-operative CSF leaks and their success rate
Gelfoam, fibrin glue, muscle graft, suturing, vinegar chloride Gelfoam, fibrin glue, muscle graft
Syre et al, 2014
13
Joseph et al, 2009
9
Gelfoam, muscle/fascia graft
Belanger et al, 2005 Epstein et al, 2009
8
Lee et al, 2014 Hannallah et al, 2008
7
Gelfoam, fascia graft, fibrin glue U-shaped microdural stables to anchor bovine pericardial graft, fibrin glue, collagen matrix, WP / LP shunt Fibrin glue
5
Primary repair
Fengbin et al, 2013 Smith et al, 1992 Present study
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Gelfoam, fibrin glue, anterior floating method Gelfoam, fascia graft Gelfoam, Durepair, Fibrin glue, DuraSeal,
Postoperative CSF leak 0/4 (0%)
None
5/13 (38%)
Suction drain, Lumbar drain None
0/9 (0%)
5/8 (63%)
None
0/5 (0%)
Complications
Re-operation
None
None
Dysphagia, headaches, would failure, sleep apnea None
Not mentioned None
Lumbar drain, CSF diversion, reexploration None
Observation, lumbar drain, Re-operation None
Anterior drain Anterior drain to gravity suction Lumbar drain None
0/7 (0%)
None
None
0/5 (0%)
None
None
3/11 (27%)
CSF cyst
5/7 (71%)
Anterior drain to gravity
0/8 (0%)
Durocutaneous fistula None
Aspiration and compression Lumbar drain, Reoperation None
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8
CSF=Cerebrospinal fluid; WP=Wound peritoneal; LP=Lumbar peritoneal
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7
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5
Postoperative drain Lumbar drain
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Intra-operative Repair method
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Lei et al, 2012
Number of intra-op CSF leak 4
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Study
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HIGHLIGHTS 1. Cerebrospinal fluid leak during anterior cervical spine surgery is a known potential complication, and must be treated in order to prevent additional
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complications, including dysphagia, airway compromise, headaches, failed fusion, poor wound healing, or even meningitis
2. Higher rates of CSF leak during anterior cervical spine surgery are associated
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with OPLL, dural ectasia in Neurofibromatosis type 1 or Marfan’s disease, intradural disk herniation, and compressive osteophytes.
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3. Herein, we present a series of 8 patients who underwent anterior cervical spinal surgery and experienced an intra-operative CSF leak with either a large gap between dural edges or with friable or frail dural edges that could not be primarily repaired.
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4. We employed a unique combination therapy, including intentional draining of CSF to a low pressure to keep the area semi dry, patching with dural substitute, sealant agent, followed by a specially shaped structural allograft that allowed for
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expansion of the patch materials while still providing adequate spinal stability. Then a locally positioned drainage bag under passive gravity drainage was placed
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at shoulder level.
5. In all 8 patients, there were no cases of wound dehiscence or CSF leak using this strategy, no evidence of cord compression or neurologic deficits. No meningitis or persistent headaches were reported, and there was no need for lumbar drain placement at any time post-operatively.
S1878-8750(16)00093-0
DOI:
10.1016/j.wneu.2016.01.014
Reference:
WNEU 3611
To appear in:
World Neurosurgery
Received Date: 23 November 2015 Revised Date:
11 January 2016
Accepted Date: 11 January 2016
Please cite this article as: Mitchell BD, Verla T, Reddy D, Winnegan L, Omeis I, Reliable Intraoperative Repair Nuances of CSF Leak in Anterior Cervical Spine Surgery and Review of The Literature, World Neurosurgery (2016), doi: 10.1016/j.wneu.2016.01.014. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Reliable Intraoperative Repair Nuances of CSF Leak in Anterior Cervical Spine Surgery and Review of The Literature
Winnegan RN1, Ibrahim Omeis MD1
Department of Neurosurgery, Baylor College of Medicine, 7200 Cambridge St,
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Bartley D. Mitchell MD PhD1, Terence Verla, MD, MPH1, Duemani Reddy MD PhD1, Lona
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Suite 9A, Houston, TX 77030
Corresponding Author:
Ibrahim Omeis MD
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Department of Neurosurgery Baylor College of Medicine
7200 Cambridge Street, suite 9A Houston, TX 77030
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Email: [email protected]
Key words: Anterior cervical spine; CSF leak; direct repair. Abbreviations: ACDF=Anterior cervical discectomy and fusion; AI=anterior instrumentation; CSF=Cerebrospinal fluid; NF=Neurofibromatosis; OPLL=Ossification of Posterior Longitudinal Ligament; PIF=posterior instrumented fusion; HLOS=Hospital length of stay; F/U=Follow up duration; WP=Wound peritoneal; LP=Lumbar peritoneal
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INTRODUCTION Cerebrospinal fluid leak during anterior cervical spine surgery is a known potential complication, and must be treated in order to prevent additional complications, including
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dysphagia, airway compromise, headaches, failed fusion, poor wound healing, or even
meningitis1-5. Although the overall incidence of CSF leak during anterior cervical spine surgery has been reported to occur in 0.2 to 1% of cases of Anterior Cervical Discectomy and Fusion
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(ACDF) or degenerative cervical spine operations6-8, there are conditions that may allow for higher rates of CSF leak during anterior cervical spine surgery, including ossification of the
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posterior longitudinal ligament (OPLL), dural ectasia in Neurofibromatosis type 1 or Marfan’s disease, intradural disk herniation, and compressive osteophytes6,7,9-13. In these cases, the dura is often tenuous at best or directly attached to the posterior longitudinal ligament, and frequently does not lend itself to primary closure, given that large portions of the dura may be missing
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altogether. There are multiple methods for treating cerebrospinal fluid leaks encountered during anterior cervical spine surgery, including primary dural closure (in cases where the dural edges are able to be sutured), fibrin glue, gel-foam, fat and fascial grafts, muscle grafts, blood patch,
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lumbar drainage, and post-operative bed positioning, and most of these methods are described in combination2,8,11,13-20 These methods have been found to be successful in many cases,
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particularly with otherwise uncomplicated anterior cervical spine surgery for ACDF, OPLL, or degenerative cervical spine. However, in cases with particularly expansive durotomies or with friable dural tissue, an alternative solution is often necessary. Herein, we present a series of 8 patients who underwent anterior cervical spinal surgery
and experienced an intra-operative CSF leak with either a large gap between dural edges or with friable or frail dural edges that could not be primarily repaired. We employed a unique
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combination therapy in the management of the CSF leak while avoiding associated complications and post-operative symptoms. Likewise, extensive review of the literature was performed to identify the different methods of treating anterior CSF leak and to compare the risk
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of failure with our method.
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METHODS In this retrospective case series, patients were included if they had: 1) undergone an anterior cervical spine surgery for one of the following conditions considered high-risk for
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durotomy and CSF leak: OPLL, Neurofibromatosis with dural ectasia, intradural disc
herniations, and compressive osteophytes, and 2) experienced a CSF leak that was recognized and treated intra-operatively. Eight patients met these criteria from this single-institution series.
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Charts were reviewed in these cases, and the methods used for repair of the CSF leak/durotomy were reviewed, along with patient outcomes and any complications that were potentially related
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to the surgical procedure. Improvement in functional outcomes was evaluated by comparing the pre and post-operative Visual Analogue Scale (VAS) and the Neck Disability Index (NDI). In each case, the same technique was used when a large CSF leak was encountered without the
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possibility of primary repair:
Intraoperative Nuances employed for dural repair: 1) Intentional drainage of CSF was performed using a cottonoid and a very low pressure
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suction to the point whereby no fluid was seen in the surgical field with the patient in a trendelenburg position, in order to keep the area around the dural defect as dry as
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possible.
2) Placement of a dural substitute, Durepair (Medtronic; Minneapolis, Minnesota), large enough to cover the entire exposed area of dura and if possible to tuck it under the adjacent bones.
3) Dry the exposed surface of the Durepair with a cottonoid and light suction.
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4) Apply DuraSeal (Covidien/Medtronic; Minneapolis, Minnesota) over the area of the dural defect with Durepair onlays, while taking care not to fill the entire expanse of the surgical cavity, given the possibility of expansion of the product, with possible dural and
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spinal cord compression.
5) Use a high-speed drill with a cutting burr to shave and shape the interbody implant on the side that will be facing towards the dura, such that it overlaps approximately the anterior
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half of the end-plate surface area.
6) Place the implant and secure the construct with an anterior titanium plating system into
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the vertebral body above and below the repair/implant site. Figure 1, part D shows the contoured implant secured with anterior instrumentation.
7) Place a tunneled percutaneous passive drain system connected to a drain bag. The catheter was placed in the surgical bed to prevent fluid accumulation/hematoma and not
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to divert CSF. The drain usually stays in place for 2 days.
All patients were allowed to sit up 3-4 hours post-operatively, and were encouraged to
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ambulate and mobilize on post-operative day 1. When the drains were removed, a nylon suture was placed at the exit site in all cases. Patients were then monitored for signs of intracranial
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hypotension, neck swelling, airway compromise, dysphagia, or meningitic symptoms, both during their hospitalization time and in the ambulatory follow-up.
Cases examples: Case 1
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80-year-old female presented with difficulty walking and weakness. Imaging studies revealed a significant C5-6 disk herniation with cord compression (Figure 1), and surgery was offered to her. Intra-operatively, there was a large defect in the dura at the C5-6 level that could
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not be repaired primarily with sutures only. The dura was opened intentionally to resect the intradural disc herniation. Given the disc was adherent to the ventral surface of the dura, it was resected with the herniated disc fragment leaving a wide opening in the dura. The dural opening
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was repaired as described above (Figure 2), with no subsequent leak. Post-operatively, there was no further drainage from the site noted. Her pain has improved as well as her functional
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mobility, and she was discharged home in good condition on post-operative day 3. Case 2
A 29 year-old man with a history of neurofibromatosis 1 and multiple neurofibromas presented with signs and symptoms of cervical myelopathy and pain radiating down his right
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upper extremity with weakness in his right upper extremity and difficulty walking for 2 weeks. CT and MRI of his cervical spine showed severe kyphotic deformity primarily at C5, with near complete subluxation and severe spinal cord compression, along with extensive dural ectasia
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(Figure 3). He underwent anterior decompression with C5 and 6 complete corpectomy with partial C4 and C7 corpectomies, with anterior and posterior instrumentation. During the anterior
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portion of the decompression, due to the dural ectasia, he had an almost non-existent dura with significant CSF leak, requiring repair as described in this paper. However, given the extent of the anterior decompression, we used an expandable cage, with a smaller diameter than we usually use to leave a space for the dural substitute/Sealant agent to expand and not cause any cord compression. Despite this, the patient did well and was able to ambulate freely post-
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operatively. He was discharged to an acute rehabilitation facility with no evidence of wound
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drainage or dehiscence. He had no evidence of dysphagia.
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RESULTS In this series, all 8 patients experienced an intra-operative CSF leak while undergoing anterior cervical spine surgery involving bony decompression with partial or complete
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corpectomy. All of the patient cases presented in this series were known to have risk factors for durotomy (NF1 with dural ectasia, OPLL, intradural disc herniation, or prominent posterior endplate osteophytes), and each had a durotomy that was not able to be closed primarily,
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necessitating an implementation of alternative strategy for prevention of CSF leak.
Table 1 shows the patient characteristics seen in this series. The patients were evenly divided by
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gender, and the age range was 30-80 years. The mean follow up duration was 31.8 months (Table 1). OPLL accounted for half the cases, the other half consisting of NF1, intradural disc herniation, and one severely ossified posterior disc osteophyte. The dural defect ranged from the size of the disc herniation of approximately 1cm to a large defect of approximately 3-4cm with
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paper-thin, floppy dural openings as in the case of the NF-1 patient. All of the patients underwent intraoperative treatment for their CSF leak as described above in the methods section. In this series using this repair protocol, there were no further CSF leaks, nor any associated
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complications typically associated with persistent cervical spine area CSF leaks. Despite draining CSF using low suction with the intradural compartment insulated with a cottonoid to achieve a
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dry surgical field, there were no complaints of headaches, nausea, vomiting or any other symptoms associated with intracranial hypotension post-operatively. All patients were monitored closely throughout their hospital stay, and all drains were removed by post-operative day 2. In the post-operative period, all patients received antibiotics until the drains were
removed. They also participated in physical therapy. The hospital length of stay ranged from 3 days (one level ACDF) to 7 days (three level Corpectomy with posterior instrumented fusion).
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By 6 months post-operatively, none of the patients had any documented complications suggesting persistent CSF leak, infections or wound breakdown. Overall, there was a significant improvement in the mean VAS scores (pre-op: 8.9 vs post-op: 1.9, p<0.0001) and NDI scores
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(pre-op: 29.4 vs post-op: 7.3, p<0.0001). Figure 4 shows the changes in the VAS and NDI scores
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following surgery utilizing the closure technique described in this study.
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DISCUSSION Anterior cervical spine surgery is a widely used surgical operation in patients with symptomatic cord compression or foraminal stenosis secondary to a variety of cervical
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pathologies including canal stenosis, myelopathy or instability21-23. Despite the success rate, there are some presenting pathologies such as OPLL, intradural disc herniation and compressive osteophyte whereby the dura mater becomes calcified and adheres to the posterior longitudinal
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ligament, increasing the risk for durotomy during anterior decompression. With the high
incidence of CSF leak with OPLL6,7,11,17,24, and with studies showing as high as 13.7% increased
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risk of CSF leak with OPLL during anterior cervical spine surgery13, we sought to describe a successful and reliable strategy for intra-operative repair of CSF leak to improve clinical outcome while reducing associated morbidity.
Each of the cases in this series was considered high risk for CSF leak because of pre-
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existing conditions such as OPLL, Neurofibromatosis with dural ectasia, intradural disk herniation, and large compressive osteophytes. In each case described, the durotomy was substantial, and illustrates the need for a reliable method of dural repair and prevention of further
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CSF leak, while still achieving the goals of the surgery, mainly decompressing the spinal cord and providing structural stability. There were no intra-operative or post-operative neurologic
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complications within 6 months in all the patients involved. No patient developed pseudarthrosis at last follow-up. Using the methods described here, we were able to achieve complete prevention of CSF leak in all cases, although the total number of cases is admittedly small. It is important to note that in each of these cases, the structural allograft used was shaped prior to implantation in order to allow more room between the structural graft and the patch materials, while still providing adequate spinal stability. This is crucial in cervical spine surgery given the
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anticipated expansion of the materials used in this algorithm, including gelfoam and fibrin glue. Without adding this extra space, it is possible for expansion of the dural repair products with compression of the spinal cord, causing severe progressing neurologic deficits, which has been
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previously reported in studies19,25.
In all of the cases described in this series, the type of Dura-Seal (Integra) used was
formulated for spinal use. This formulation differs from the cranial formulation in that although
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it does expand, it does not expand nearly to the same degree as the cranial type. Nevertheless, in performing this particular intra-operative CSF leak control method, we recommend allowing
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extra room for allograft expansion. Careful observation should be made in the immediate postoperative phase, with a high index of suspicion for graft-expansion in the event of rapid declining neurologic status. In addition, we intentionally drained CSF intraoperatively using a cottonoid and low pressure suction to create a relatively dry environment for the dural substitute
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to adhere to the dura once the sealant agent was applied. This avoids re-accumulation of CSF, which could potentially cause dehiscence of the patch. Several studies have reported different strategies for the management of intra-operative
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CSF leak during anterior cervical spine surgery with varying success rates. Intra-operative repair can be achieved using gelatin foam, fibrin glue, dural grafts, DuraSeal, muscle grafts or any
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combination of these2,8,13,15-20. Post-operatively, lumbar CSF drains and lumbar/wound-peritoneal shunts have been utilized as a preventive measures to reduce CSF leaks6,8,16,17,26-28. In a series by Hannallah et al13, 20 patients were identified with intra-operative CSF leak. Five patients with accessible dural tears underwent intraoperative repair with the use of anterior cervical drains kept on gravity-drainage and discontinued on post-operative day 2. Another series of 13 patients by Syre et al8 reported successful intraoperative repair of 8 CSF leaks using a combination of
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gelfoam, fibrin glue, muscle graft and Duragen, without need for additional treatment post-op. The remaining 5 patients required lumbar drains, re-exploration and shunts for persistent CSF leak. In addition to intraoperative repair, several studies have utilized placement of lumbar
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drains and lumbar/wound-peritoneal shunts to achieve satisfactory outcomes8,16,17,28-30. Table 2 shows a summary of clinical cases with intra-operative repair of CSF leaks with or without the placement of lumbar drains or shunts.8,13,16,17,20,28,31-33
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There may be no one single method for repairing large durotomies, and certainly the method described in our series may not work on all leaks. The method described here does not
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offer an entirely new concept either, rather it uses a particular combination of repair strategies combined with the use of intentional drainage of CSF to the point it becomes dry at the durotomy site followed by direct patching of the defect with a sealant. We believe extra drainage of CSF prior to the initiation of patching may give some time for the patch to adhere to the dura and
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prevent dehiscence by applying the sealant agent to cover it and provide some pressure. Then, utilizing a passive drain (drain to gravity, not suction) allowed for drainage of any excess fluid that may accumulate. This presumably may prevent complications caused by fluid compression
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of the trachea (respiratory distress), esophagus (dysphagia), or other structures, while preventing active siphoning of fluid, thus preventing the dural repair from truly sealing. This is similar to
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Hannallah et al13 in their approach to accessible dural tears with the use of gravity drainage, achieving satisfactory clinical outcomes. Furthermore, our protocol does not utilize lumbar drains and lumbar/wound-peritoneal shunts which can add its own set of complications11. In our study, there was a significant improvement in functional outcomes (VAS and NDI)
at 6 weeks following surgery and there was no post-operative complication in all eight patients. This illustrates the safety and reliability of our strategy at intraoperative management of CSF
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leaks in circumstances whereby the extent of the dural tear is not amendable to primary closure
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techniques.
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CONCLUSION Overall, this method was reliable and reproducible in 8 consecutive cases, with straightforward strategies and nuances that can be implemented readily during the encounter of
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an anterior cervical spine dural opening whether it was intentional or not. Although the
development of more methods for repair of large durotomies is certainly warranted, this method
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persistent CSF leaks from anterior spine surgery.
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may be a useful tool in any surgeon’s armamentarium to avoid morbidity associated with
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lacerations in patients with ossification of the posterior longitudinal ligament. Surg Neurol 2009;72:630-4; discussion 4. Choi S, Lee SH, Lee JY, et al. Factors affecting prognosis of patients who underwent
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Results of anterior cervical decompression and arthrodesis in sixty-one North American patients. The Journal of bone and joint surgery. American volume 2005;87:610-5. Lee SE, Chung CK, Jahng TA, et al. Dural tear and resultant cerebrospinal fluid leaks
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FIGURE LEGEND: Figure 1: (Case1) 80y/o F with significant C5-6 disk herniation with cord compression presenting with difficulty walking and weakness. She underwent C5-6 ACDF.
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(A) Sagittal, (B) Axial views of CT scan demonstrating the intradural calcified disc herniation. (C) Intra-operative X-Ray showing the position of the graft (arrow) shortened to allow expansion of the sealant agent.
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(D) Lateral XR-Ray with drawings showing the graft and materials used to seal the dural defect. (The graft (red) is reduced in size to allow for the sealant agent (Blue) and dural substitute
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(Brown) to expand without causing cord compression. (White) is space left between the graft and the sealant agent.
Figure 2: Intraoperative images showing
(A) Intradural calcified disc fragment (arrow: dural edge; asterisk: large intradural calcified
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fragment).
(B) Exposure of the spinal cord after resection of the disc fragment (C) Placement of a dry dural substitute, and (D) sealant agent.
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Figure 3: (Case 2) 29y/o M with history of NF-1, presents with severe kyphotic deformity, with near complete subluxation and severe spinal cord compression, along with extensive dural
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ectasia. He presented with cervical myelopathy and radiating pain down his upper extremity with weakness. He underwent cervical corpectomies followed by placement of expandable cage and C3 to T1 anterior instrumentation. Sagittal CT (A), MRI (B) showing the extensive pathology of neurofibroma, dural ectasia and subluxation. (C) post-op CT scan showing reconstruction of the anterior spine.
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Figure 4: Significant improvement in the pre- and post-operative VAS and NDI scores in patients undergoing intra-operative repair of CSF leak using the technique described in this
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study.
TABLE LEGEND
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Table 1: Characteristic summary of the 8 patients included in the series
Table 2: Clinical studies describing the management of intra-operative CSF leaks and their
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success rate
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Table 1: Characteristic summary of the 8 patients included in the series Gender
Presenting symptoms
Preoperative diagnosis
Level of surgery
1
80
F
29
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Intradural disc herniation NF-1 with neurofibroma and dural ectasia
C5-6 ACDF
2
Weakness, walking difficulty Radiating neck pain
3
48
F
OPLL
4
70
M
Radiating neck pain, walking difficulty Walking Difficulty
5
54
M
6
47
Radiating neck pain Radiating neck pain, falls, walking difficulty
Compressive osteophyte OPLL and Compressive osteophyte
7
61
8
48
Duration of post-op wound drainage 2
Post-op complications (within 6mths)
HLOS (days)
F/U (mths)
Disposition
None
3
13
Home
None
2
None
7
42
Acute Rehab – Symptom resolution at last f/u
None
2
None
5
22
Home
None
2
4
51
Acute Rehab - Symptom resolution at last f/u Home
None
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OPLL
C4-7 corpectomies with C3-T1 anterior instrumentatio n C5 C with C4-6 AI followed by PIF C4-5 ACDF
Postop CSF leak None
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Age
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Case #
AC C
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C5-6 None 2 None 3 32 ACDF F C3-4 ACDF None 2 None 5 42 Patial C3, complete C4 C with C3-5 AI followed by PIF M Radiating OPLL and C4 C with None 2 None 5 28 neck pain, Compressive C3-5 AI with falls, osteophyte PIF F Numbness, OPLL C4-5 C None 2 None 6 25 weakness With C3-6 AI followed by PIF AI=anterior instrumentation; C=corpectomy; PIF=posterior instrumented fusion. OPLL=Ossification of Posterior Longitudinal Ligament; ACDF=Anterior cervical discectomy and fusion; NF=Neurofibromatosis; HLOS=Hospital Length of stay. F/U=Follow up duration
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ACCEPTED MANUSCRIPT
Table 2: Clinical Studies describing the management of intra-operative CSF leaks and their success rate
Gelfoam, fibrin glue, muscle graft, suturing, vinegar chloride Gelfoam, fibrin glue, muscle graft
Syre et al, 2014
13
Joseph et al, 2009
9
Gelfoam, muscle/fascia graft
Belanger et al, 2005 Epstein et al, 2009
8
Lee et al, 2014 Hannallah et al, 2008
7
Gelfoam, fascia graft, fibrin glue U-shaped microdural stables to anchor bovine pericardial graft, fibrin glue, collagen matrix, WP / LP shunt Fibrin glue
5
Primary repair
Fengbin et al, 2013 Smith et al, 1992 Present study
11
Gelfoam, fibrin glue, anterior floating method Gelfoam, fascia graft Gelfoam, Durepair, Fibrin glue, DuraSeal,
Postoperative CSF leak 0/4 (0%)
None
5/13 (38%)
Suction drain, Lumbar drain None
0/9 (0%)
5/8 (63%)
None
0/5 (0%)
Complications
Re-operation
None
None
Dysphagia, headaches, would failure, sleep apnea None
Not mentioned None
Lumbar drain, CSF diversion, reexploration None
Observation, lumbar drain, Re-operation None
Anterior drain Anterior drain to gravity suction Lumbar drain None
0/7 (0%)
None
None
0/5 (0%)
None
None
3/11 (27%)
CSF cyst
5/7 (71%)
Anterior drain to gravity
0/8 (0%)
Durocutaneous fistula None
Aspiration and compression Lumbar drain, Reoperation None
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8
CSF=Cerebrospinal fluid; WP=Wound peritoneal; LP=Lumbar peritoneal
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7
AC C
5
Postoperative drain Lumbar drain
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Intra-operative Repair method
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Lei et al, 2012
Number of intra-op CSF leak 4
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Study
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AC C
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AC C
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AC C
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HIGHLIGHTS 1. Cerebrospinal fluid leak during anterior cervical spine surgery is a known potential complication, and must be treated in order to prevent additional
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complications, including dysphagia, airway compromise, headaches, failed fusion, poor wound healing, or even meningitis
2. Higher rates of CSF leak during anterior cervical spine surgery are associated
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with OPLL, dural ectasia in Neurofibromatosis type 1 or Marfan’s disease, intradural disk herniation, and compressive osteophytes.
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3. Herein, we present a series of 8 patients who underwent anterior cervical spinal surgery and experienced an intra-operative CSF leak with either a large gap between dural edges or with friable or frail dural edges that could not be primarily repaired.
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4. We employed a unique combination therapy, including intentional draining of CSF to a low pressure to keep the area semi dry, patching with dural substitute, sealant agent, followed by a specially shaped structural allograft that allowed for
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expansion of the patch materials while still providing adequate spinal stability. Then a locally positioned drainage bag under passive gravity drainage was placed
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at shoulder level.
5. In all 8 patients, there were no cases of wound dehiscence or CSF leak using this strategy, no evidence of cord compression or neurologic deficits. No meningitis or persistent headaches were reported, and there was no need for lumbar drain placement at any time post-operatively.