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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 3  |  Issue : 2  |  Page : 254-257

A rare case of spontaneous pseudomeningocele with lytic spondylolisthesis


Department of spine, Stavya Spine Hospital and Research Institute, Ahmedabad, Gujarat, India

Date of Submission26-Mar-2019
Date of Decision03-May-2019
Date of Acceptance13-Aug-2019
Date of Web Publication13-Jul-2020

Correspondence Address:
Dr. Devanand Degulmadi
Department of spine, Stavya Spine Hospital and Research Institute, Mithakali, Ahmedabad, Gujarat.
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/isj.isj_20_19

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  Abstract 

Pseudomeningocele is an extra dural collection of cerebrospinal fluid arising from dural defect secondary to incidental durotomy, trauma or congenital. Lytic spondylolisthesis as a cause of dural defect leading to pseudomeningocele has been rarely reported. Pars defect in lytic spondylolisthesis has been documented to have highly organized collagen bundles and calcified fibro cartilaginous enthesis which probably leads to dural injury. We report a case of 56 year old female with grade 2 lytic spondylolisthesis at L4-L5 associated with spontaneous pseudomeningocele. During surgery CSF leak was noted with dural defect and herniated rootlets entangled in facet capsule. Underlying pathogenesis, imaging, surgical findings and management are discussed.

Keywords: Fibrocartilaginous enthesis, lytic spondylolisthesis, spontaneous pseudomeningocele


How to cite this article:
Bang PB, Dave BR, Degulmadi D, Krishnan A. A rare case of spontaneous pseudomeningocele with lytic spondylolisthesis. Indian Spine J 2020;3:254-7

How to cite this URL:
Bang PB, Dave BR, Degulmadi D, Krishnan A. A rare case of spontaneous pseudomeningocele with lytic spondylolisthesis. Indian Spine J [serial online] 2020 [cited 2020 Oct 30];3:254-7. Available from: https://www.isjonline.com/text.asp?2020/3/2/254/289647




  Introduction Top


Dural defects resulting in abnormal extradural cerebro spinal fluid (CSF) collection (pseudomeningocele) have been reported in the literature that may be congenital, iatrogenic, or secondary to trauma.[1] A lytic portion of the spondylolisthesis as a cause of pseudomeningocele has been reported rarely. Pars defect in lytic spondylolisthesis has been documented to have highly organized collagen bundles and calcified fibrocartilaginous enthesis.[2] Such calcified spurs on contact with dura pose a risk of dural injury. We present a case of a pseudomeningocele associated with lytic spondylolisthesis, the underlying possible causes of such pathology, and its surgical management.


  Case Report Top


A 56-year-old woman presented with complaints of low back pain since a few years and acute left leg pain for three weeks. Leg pain increased on sitting, standing, and walking, limiting her walking distance to 100 meters. She described this pain as an electric shock-like sensation that radiated along the medial part of the left leg corresponding to L4 dermatome. There was no history of trauma, previous lumbar surgery, or any injections in her back. On examination, there was a palpable step at the level of L4-5, and neurology was normal. Standing X-rays of lumbosacral spine showed a grade 2 lytic spondylolisthesis at L4-5 level [Figure 1]. T2-weighted magnetic resonance imaging (MRI) showed a hyperintense signal in interspinal and both paraspinal areas at the level of L4-5, suggestive of fluid collection [Figure 2]A and B. This abnormal collection of fluid was seen to be in continuation with the spinal cord on myelogram, indicative of CSF [Figure 2]C.
Figure 1: Anteroposterior view of lumbar spine (A), dynamic flexion and extension views of lumbar spine showing grade 2 lytic listhesis (B)

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Figure 2: T2 sagittal magnetic resonance image (MRI) showing hyperintense signal in interspinal area of L4-L5 (Black arrow) (A), T2 axial image showing hyperintense signal in both paraspinal areas (B), MRI myelogram showing continuation of fluid in interspinal area to spinal cord (Black arrow) (C)

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The patient was planned for L4-5 fusion surgery. During exposure, the fluid started to leak below the lumbodorsal fascia. The fluid was identified to be clear in nature, consistent with CSF. After completion of the exposure and laminectomy, the dural defect was identified close to left-exiting L4 nerve root on the dorsolateral aspect of the dural sac. Multiple rootlets were found to be lying outside the dura. Three rootlets were found to be entangled with facet capsule that required careful dissection. Two of three were repositioned in dural sac, whereas one was excised as it could not be retrieved. The dural defect was identified and repaired with continuous suture with fat graft interposition. The routine procedure of interbody fusion was performed, and the rest of the surgery was uneventful [Figure 3]A and B. Watertight closure was performed with positive pressure drain. Postoperative neurology was normal and leg pain was completely relieved. Adequate supportive treatment with intravenous antibiotics, oral acetazolamide, head low position, and oral fluids was given for the first two days. The patient was mobilized on the third postoperative day. Dressing was dry after 72 h with drain less than 50 mL. As per the protocol at our institute, the drain was removed as it was less than 50 mL over 24 h. MRI screening on the 15th day after suture removal showed no collection [Figure 3]C. The patient neurologically improved and was doing well at 18-month follow-up [Figure 4].
Figure 3: Postoperative anteroposterior view of lumbar spine (A), postoperative lateral view of lumbar spine (B), postoperative T2 magnetic resonance imaging sagittal view at 15 days showing no collection (C)

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Figure 4: (A, B) Postoperative T2 sagittal and axial image at 18-month follow-up with no collection

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  Discussion Top


Pseudomeningocele is classified as congenital, traumatic, and iatrogenic.[1] It can also occur in conditions such as spina bifida occulta, neurofibromatosis, and Marfan syndrome where the dura is prone to rupture because of ectasia.[3],[4] The most common cause is iatrogenic where there is an inadvertent durotomy during spinal decompression. The exact incidence of pseudomeningocele is difficult to estimate; a majority of them remain asymptomatic and go unnoticed.[5] It is more commonly seen after lumbar surgeries because of high intrathecal pressure and a large number of surgeries performed in this region. Other rare causes of pseudomeningocele include epidural injections and use of anti-adhesive gels in lumbar spine surgery.[6]

Spontaneous pseudomeningocele is a rare type where there is no significant history suggestive of the etiology. Its association with lytic spondylolisthesis needs further emphasis and understanding of the basic pathogenesis. Lytic listhesis is associated with a well-organized collagenous and fibrocartilaginous enthesis with calcifications and bony spurs at the pars defect.[2] There is also an associated thinning of the dura under the lysis defect. Dura is prone to injury in such cases, leading to dural defect and a spontaneous CSF leak.

Novais et al.[7] have published a case report on spontaneous pseudomeningocele where they described its association with lytic spondylolisthesis. Although our case has the same preoperative imaging, the clinical and intraoperative findings are distinct. During exploration, the typical encapsulation seen in pseudomeningocele was absent, and the leak started immediately as the lumbodorsal fascia was opened. This probably indicates that the event was acute in nature. The clinical finding of electric shock-like pain for only three weeks over a chronic low back pain for the last few years also confirms the same. An intraoperative finding of rootlets lying outside the defect with three rootlets entrapped in the facet capsule and its disappearance after the surgery when these rootlets were relieved, further corroborates with the aforementioned clinical scenario.

Very careful handling of neurological structures is must in such cases. Intraoperative repair of the defect with 5-0 or 6-0 continuous sutures with nonabsorbable material is recommended. Anti-adhesive gels should be discouraged as they prevent fibrocyte migration and healing.[6] A fat, fascia, or muscle graft can be used to augment the efficacy of the repair. Some also recommend the use of fibrin glue to prevent any further micro-leaks. A watertight closure over a drain in positive mode and Trendelenburg position in the ward are practiced to prevent further complications. CSF pressure lowering drugs such as acetazolamide can be started. If the postoperative wound starts leaking, it is recommended to reexplore.[8]


  Conclusion Top


Spontaneous pseudomeningocele with lytic spondylolisthesis is a rare entity and needs careful evaluation. All patients with lytic spondylolisthesis should be evaluated for hyperintense signals in the interspinal and paraspinal area. Acute leg pain may indicate an event of spontaneous dural injury or root entrapment, which should be managed cautiously.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Solomon P, Sekharappa V, Krishnan V, David KS. Spontaneous resolution of postoperative lumbar pseudomeningoceles: A report of four cases. Indian J Orthop 2013;47:417-21.  Back to cited text no. 1
  [Full text]  
2.
Boszczyk BM, Boszczyk AA, Boos W, Korge A, Mayer HM, Putz R, et al. An immunohistochemical study of the tissue bridging adult spondylolytic defects—The presence and significance of fibrocartilaginous entheses. Eur Spine J 2006;15:965-71.  Back to cited text no. 2
    
3.
Arnold PM, Teuber J. Marfan syndrome and symptomatic sacral cyst: Report of two cases. J Spinal Cord Med 2013;36: 499-503.  Back to cited text no. 3
    
4.
Banno T, Ohishi T, Suzuki D, Honda Y, Kobayashi S, Matsuyama Y. Traumatic sacral pseudomeningocele with spina bifida occulta. J Neurosurg Spine 2012;16:78-81.  Back to cited text no. 4
    
5.
Hawk MW, Kim KD. Review of spinal pseudomeningoceles and cerebrospinal fluid fistulas. Neurosurg Focus 2000;9:e5.  Back to cited text no. 5
    
6.
Kuhn J, Hofmann B, Knitelius HO, Coenen HH, Bewermeyer H. Bilateral subdural haematomata and lumbar pseudomeningocele due to a chronic leakage of liquor cerebrospinalis after a lumbar discectomy with the application of ADCON-L gel. J Neurol Neurosurg Psychiatry 2005;76:1031-3.  Back to cited text no. 6
    
7.
Novais G, Ratilal B, Pappamikail L, Branco P, Reis N. Spontaneous pseudomeningocele associated with lumbar spondylolisthesis: A case report and review of the literature. Surg Neurol Int 2017;8:221.  Back to cited text no. 7
  [Full text]  
8.
Eismont FJ, Wiesel SW, Rothman RH. Treatment of dural tears associated with spinal surgery. J Bone Joint Surg Am 1981;63:1132-6.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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