Neurology India
menu-bar5 Open access journal indexed with Index Medicus
  Users online: 4480  
 Home | Login 
About Editorial board Articlesmenu-bullet NSI Publicationsmenu-bullet Search Instructions Online Submission Subscribe Videos Etcetera Contact
  Navigate Here 
  » Next article
  » Previous article 
  » Table of Contents
 Resource Links
  »  Similar in PUBMED
 »  Search Pubmed for
 »  Search in Google Scholar for
 »Related articles
  »  Article in PDF (105 KB)
  »  Citation Manager
  »  Access Statistics
  »  Reader Comments
  »  Email Alert *
  »  Add to My List *
* Registration required (free)  

  In this Article
 »  Abstract
 »  Introduction
 »  Case Report
 »  Discussion
 »  References

 Article Access Statistics
    PDF Downloaded144    
    Comments [Add]    
    Cited by others 5    

Recommend this journal

Year : 2003  |  Volume : 51  |  Issue : 3  |  Page : 390-391

Delayed post-surgical development of dural arteriovenous fistula after cervical meningocele repair

Department of Neurosurgery, The Royal Victoria Hospital, Grosvenor Road, Belfast BT12 6BA,

Correspondence Address:
Department of Neurosurgery, The Royal Victoria Hospital, Belfast BT12 6BA
[email protected]

  »  Abstract

A 34-year-old female patient presented with an intracranial subarachnoid hemorrhage and was found to have a dural arteriovenous fistula at the site of previous cervical meningocele repair. Subsequent occlusion was achieved with endovascular embolization. To our knowledge, the phenomenon of the development of a spinal dural fistula at the site of a meningocele repair has not been recorded before.

How to cite this article:
Flannery T, Tan M H, Flynn P, Choudhari K A. Delayed post-surgical development of dural arteriovenous fistula after cervical meningocele repair . Neurol India 2003;51:390-1

How to cite this URL:
Flannery T, Tan M H, Flynn P, Choudhari K A. Delayed post-surgical development of dural arteriovenous fistula after cervical meningocele repair . Neurol India [serial online] 2003 [cited 2021 Mar 7];51:390-1. Available from:

   »   Introduction Top

A spinal dural arteriovenous fistula (SDAVF) is a direct communication of artery to vein located at the dural covering of the spinal nerve root.[1] There is usually a single artery or rarely, multiple feeding arteries connected to a single radicular vein, which then drains to the perimedullary veins.[2] Patients usually present with myelopathic symptoms which are thought to arise secondary to venous hypertension and hypoperfusion of the spinal cord.[3] Rarely, SDAVFs may present with subarachnoid hemorrhage.[4],[5] We report a case of a patient who had a cervical meningocele repair as an infant and presented with subarachnoid hemorrhage secondary to a cervical SDAVF thirty-three years later.

   »   Case Report Top

A 34-year-old female with a previous history of a cervical meningocele repaired at the first two cervical levels (C1-2) as an infant, was admitted with a sudden onset of occipital headache, loss of consciousness lasting 15 minutes, vomiting and an episode of urinary incontinence. On clinical examination, she was conscious, alert, orientated with neck stiffness. She was also noted to have a scar at the back of her neck, which was subsequently found to be the site of her cervical meningocele repair in infancy. A computerized tomography (CT) scan of the brain revealed subarachnoid hemorrhage with intraventricular extension. There was associated ventriculomegaly but it was felt to represent chronic arrested hydrocephalus. Transfemoral four-vessel cerebral angiography revealed a small extracranial arteriovenous fistula in the midline at the level of the spinous process of the first cervical vertebra (C1) [Figure - 1].
It was supplied by the cervical branches of the right and left vertebral arteries. Three days later, she underwent endovascular embolization of the arteriovenous malformation (AVM) with cyanoacrylate glue. The feeding branch from the left vertebral artery was successfully obliterated while the right could only be partially embolized [Figure - 2]. However, almost complete obliteration of the fistula was achieved. The patient tolerated the procedure well and was discharged without neurological deficits.

   »   Discussion  Top

SDAVFs, the most common spinal AVMs, are being increasingly recognized since the advent of superselective spinal angiography.[6] The typical angiogram appearance is that of a slow-flow arteriovenous fistula in the neural foramen draining through dilated tortuous perimedullary veins located on the surface of the spinal cord.[2] They are thought to develop following venous thrombosis of the intradural spinal veins.[4] Most SDAVFs are located in the thoracic and lumbar regions while those arising in the cervical region account for a small percentage of cases.[4] Those in the cervical spine can be fed by the branches of either the thyrocervical and costocervical trunk and/or both vertebral arteries.[7]
While spinal AVMs have been reported to occur in association with meningomyelocele,[8] no fistulous communication was evident at the time of the initial surgery in this case. Although it is possible that the occurrence of a SDAVF at the site of previous cervical meningomyelocele repair is purely coincidental, the development of dural arteriovenous fistulae following previous trauma and surgery is well reported.[9],[10],[11],[12] Postoperative fibrosis may have accounted for impaired drainage of the dural venous plexus with consequent venous thrombosis of the intradural spinal veins. There is widespread agreement that thrombosis of the intradural draining veins is responsible for SDAVF formation.[13] While the origin of this patient's hemorrhage is unclear, it is possible that venous hypertension in the arterialized vein and the draining perimedullary veins may have precipitated rupture and venous hemorrhage.[14]
Although spinal vascular malformations represent less than 4% of all spinal cord masses, they are important clinical entities because they produce considerable morbidity and may even be fatal if left untreated.[15] The mortality rate associated with spinal subarachnoid hemorrhage caused by spinal vascular malformations is up to 20%.[15] Treatment of a SDAVF consists of obliteration of the artery-to-vein communication, which can be done by surgical intervention or by an embolization procedure. Embolization of SDAVFs is associated with a 60 to 90% obliteration rate; the recurrence or recanalization rate may be higher with certain agents, such as polyvinyl alcohol, than with acrylic glues.[16] Surgical ligation is often reserved for cases with embolization failure or in case of inability to safely embolize the lesion.[17]
Though spinal AVMs are known to occur in association with meningomyelocele, most SDAVFs are thought to be acquired through venous thrombosis of intradural spinal veins. We believe this is the first reported case of the development of a cervical SDAVF in a patient who previously had a cervical meningocele repair. The possible significance of previous surgery at the craniocervical junction, in a patient presenting with intracranial subarachnoid hemorrhage, needs to be highlighted. It should also be pointed out that embolization may not provide a long-term cure. Recurrence of this condition is possible and may necessitate surgical exploration. As such, long-term follow-up of the patient is recommended.

  »   References Top

1.Niimi Y, Setton A, Berenstein A. Spinal dural arteriovenous fistula draining to the anterior spinal vein: Angiographic diagnosis. Neurosurgery 1999;44:  Back to cited text no. 1    
2.999-1004.  Back to cited text no. 2    
3.Bernheim N, Poirier J, Hurth M. Arteriovenous fistula of the meninges draining into the spinal veins: A histologic study of 28 cases. Acta Neuropathol (Berl) 1983;62:103-11.  Back to cited text no. 3    
4.Berenstein A, Lasjaunias P. Spine and spinal cord vascular lesions In: Surgical Neuroangiography: Endovascular treatment of Spine and Spinal Cord lesions: Springer-Verlag, Berlin: 1992. pp. 1-109.   Back to cited text no. 4    
5.Morimoto T, Yoshida S, Basugi N. Dural arteriovenous malformations in the cervical spine presenting with subarachnoid haemorrhage: case report. Neurosurgery 1992;31:118-21.  Back to cited text no. 5    
6.Glasser R, Masson R, Mickle JP, Peters KR. Embolization of a dural arteriovenous fistula of the ventral cervical spinal canal in a nine-year-old boy. Neurosurgery 1993;33:1089-94.  Back to cited text no. 6    
7.Djindjian R. Angiography of the spinal cord. Surg Neurol 1974;2:175-85.  Back to cited text no. 7    
8.Watts C, Ryken TC. Intraspinal haemorrhage from idiopathic or structural causes In: Grossman RG, Loftus CM, editors. Principles of Neurosurgery. 2nd edn. Philadelphia: Lippincott-Raven; 1999. pp. 630-1.  Back to cited text no. 8    
9.Chatkupt S, Ruzicka PO, Lastra CR. Myelomeningocele, spinal arteriovenous malformations & epidermal naevi syndrome: a possible rare association. Dev Med Child Neurol 1993;35:737-41.  Back to cited text no. 9  [PUBMED]  
10.Bito S, Ohnishi T, Takimoto N, Sakaki S, Gohma T, Motozaki T. Dural arteriovenous fistulae found after removal of meningiomas: A case report. Neurol Surg 1978;6:397-400.  Back to cited text no. 10  [PUBMED]  
11.Dennery JM, Ignacio BS. Post-traumatic arteriovenous fistula between the external carotid arteries and the superior longitudinal sinus: Report of a case. Can J Surg 1967;10:333-6.  Back to cited text no. 11  [PUBMED]  
12.Ishii R, Ueki K, Ito J. Traumatic fistula between a lacerated middle meningeal artery and a diploic vein: Case report. J Neurosurg 1976;44:241-4.  Back to cited text no. 12  [PUBMED]  
13.Wilson CB, Cronic F. Traumatic arteriovenous fistulas involving middle meningeal vessels. JAMA 1964;188:953-7.  Back to cited text no. 13  [PUBMED]  
14.Bederson JB, Spetzler RF. Pathophysiology of type I spinal arteriovenous malformations. BNIQ 1996;12:23-32.  Back to cited text no. 14    
15.Hassler N, Thron A, Grote EH. Hemodynamics of spinal dural arteriovenous fistulae: an intraoperative study. J Neurosurg 1989;70:360-70.  Back to cited text no. 15    
16.Aminoff MJ, Logue V. The prognosis of patients with spinal vascular malformations. Brain 1974;97:211-8.  Back to cited text no. 16  [PUBMED]  
17.Nichols DA, Rufenacht DA, Jack CR, Forbes GS. Embolization of spinal dural arteriovenous fistulae with polyvinyl alcohol particles: experience in 14 patients. Am J Neuroradiol 1992;13:933-40.  Back to cited text no. 17    
18.Mourier KL, Gelbert F, Rey A, Assouline E, George B, Reizine D, Merland JJ, Cophignon J. Spinal dural arteriovenous malformations with perimedullary drainage: indication and results of surgery in 30 cases. Acta Neurochir (Wien) 1989;100:136-41.  Back to cited text no. 18  [PUBMED]  


Print this article  Email this article
Previous article Next article
Online since 20th March '04
Published by Wolters Kluwer - Medknow