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|LETTERS TO EDITOR
|Year : 2019 | Volume
| Issue : 5 | Page : 1376-1379
Spontaneous Closure of Dural Arteriovenous Fistula; A Visual Specter
Manish Beniwal1, Jitender Saini2, Sampath Somanna1, Aarthi Deepesh2, K V L N Rao1, Vikas Vazhayil1, Dwarkanath Srinivas1
1 Department of Neurosurgery, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India
2 Department of Neuroimaging and Interventional Radiology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India
|Date of Web Publication||19-Nov-2019|
Dr. Jitender Saini
Department of Neuroimaging and Interventional Radiology, National Institute of Mental Health and Neurosciences, Bengaluru - 560 029, Karnataka
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Beniwal M, Saini J, Somanna S, Deepesh A, Rao K V, Vazhayil V, Srinivas D. Spontaneous Closure of Dural Arteriovenous Fistula; A Visual Specter. Neurol India 2019;67:1376-9
|How to cite this URL:|
Beniwal M, Saini J, Somanna S, Deepesh A, Rao K V, Vazhayil V, Srinivas D. Spontaneous Closure of Dural Arteriovenous Fistula; A Visual Specter. Neurol India [serial online] 2019 [cited 2019 Dec 10];67:1376-9. Available from: http://www.neurologyindia.com/text.asp?2019/67/5/1376/271248
Dural arteriovenous fistula (DAVF) is a rare type of intracranial malformation which accounts for 10–15% of all intracranial arteriovenous malformations. They usually occur in middle-aged adults, most commonly in the 6th decade and in a female to male ratio of 1.65:1. Few cases have a benign course, but some develop spontaneous closures for unknown reasons. We report a case of bilateral dural AVF fistula cognard type II (a + b) with spontaneous closure, presenting to us with features of raised intracranial pressure and with vision loss.
A 19-year-old male presented with history of a head injury which occurred 18 months previously. Investigations revealed superior sagittal sinus and right transverse sinus thrombosis and he was started on oral anticoagulants. He took medications for 1 year and then stopped the medications on his own and was lost to follow-up.
He returned few months/years later with complaints of an occipital headache, insidious onset, dull aching associated with blurring of vision, and one episode of loss of consciousness.
He was investigated with magnetic resonance imaging (MRI) which revealed dilatation and tortuosity of both superior ophthalmic veins prominently on the right side suggestive of bilateral DAVF. Ophthalmological examination revealed his vision was 6/36 in the right eye and 6/12 in the left eye. Perimetry showed paracentral, centrocecal, and superotemporal arcuate scotoma in the right visual field with a visual field index of 63%. His left visual field also showed central and paracentral scotoma and VFI of 46% [Figure 1]a and [Figure 2]a.
|Figure 1: (a) Right showing scotoma and visual field index (VFI) of 63%, (b) Right eye showing an increase in scotomas and decrease in VFI to 21%, (c and d) Right eye fundus showing gross papilledema|
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|Figure 2: (a) visual fields and VFI in the left eye, (b) showing an increase in scotomas and decrease in VFI to 16%, (c and d) Left eye also shows gross papilledema|
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He was further evaluated with digital subtraction angiogram (DSA) which revealed bilateral DAVF [Figure 3] and [Figure 4]. He was advised for embolization of DAVF but was lost to follow-up. He came back 3 months later with an increased complaint of a headache and visual blurring. He was scheduled for embolization. When the pre-embolization DSA was performed, it showed the complete disappearance of DAVF [Figure 5].
|Figure 3: Right transverse sinus DAVF is seen with feeders from the dural branch of the meningohypophyseal trunk (MHT) of the right ICA (a), Transosseous branches of the right occipital artery, the petrous branch of the right middle meningeal artery and from the neuromeningeal trunk of the ascending pharyngeal artery (b), (c and d) Retrograde filling of the sinus with the minimal antegrade flow. Antegrade flow beyond sigmoid sinus is not seen. Reflux into the cortical veins is noted|
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|Figure 4: (a) Left transverse sinus DAVF is seen with feeders from the dural branches of the MHT of right ICA, (b) Transosseous branches of the left occipital artery and from the neuromeningeal trunk of the ascending pharyngeal artery. The DAVF also had feeder from the petrous branch of the middle meningeal artery (not shown), (c and d) There is retrograde filling of the sinus with a minimal antegrade flow. Antegrade flow beyond sigmoid sinus is not seen. Reflux into the cortical veins are also noted|
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|Figure 5: (a) Left ICA injection did not reveal filling of the transverse sinus, (b) Right external carotid injection did not reveal any evidence of transverse sinus DAVF as seen in the previous angiogram done 2 months earlier suggestive of spontaneous thrombosis of the DAVF, (c) Right ICA injection did not reveal any feeders from the MHT as seen previously feeding the transverse sinus fistula,(d) Left selective occipital artery injection, however, showed a small transosseous feeder still opacifying the left transverse sinus faintly. There was no cortical venous reflux seen. The other feeders also regressed|
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He underwent a repeat ophthalmological examination, which showed visual acuity of 6/36 in the right eye and 6/18 in left eye. Fundus revealed gross papilledema in both eyes [Figure 1]c, [Figure 1]d and [Figure 2]c, [Figure 2]d. Perimetry examination showed an increase in scotomas and a decrease in VFI in both eyes [Figure 1]b and [Figure 2]b.
Cerebrospinal fluid (CSF) manometry was done through lumbar puncture, which revealed a CSF pressure of 300 mm of water. Because of the raised intracranial pressure, he underwent Lumbar-peritoneal shunt. After the CSF diversion procedure, his headache was relieved and his vision stabilized, and patient reported no further vision blurring.
At 4 months follow-up, patient vision improved to 6/9 in the right eye and 6/12 in the left eye and resolved papilledema [Figure 6].
|Figure 6: (a) Fundus image of the right eye showing resolved papilledema, (b) Fundus image of the left eye also showed resolved papilledema|
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At 1 year follow-up, the patient underwent repeat DSA, which showed closure of DAVF [Figure 7] and [Figure 8].
|Figure 7: (a and b) Right and left ICA injections in lateral view did not reveal any feeders from the MHT as seen previously feeding the transverse sinus fistula, (c and d) Right vertebral artery injection in AP and lateral views also did not reveal any evidence of fistula|
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|Figure 8: (a and b) Right ECA injection AP and lateral planes did not reveal filling of the fistula. Note a prominent arterial channel along the wall of the right transverse and distal superior sagittal sinus. However, there was no fistulous communication or filling of the sinus, (c and d) Left external carotid injection AP and lateral views did not reveal any evidence of transverse sinus DAVF as seen in the previous angiogram done earlier suggestive of spontaneous thrombosis of the DAVF|
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Intracranial DAVF is an abnormal arterial venous connection located in the intracranial dura mater. The most common classification system used is given by Borden and Cognard., The exact etiopathogenesis of DAVF has not been elucidated till now. It is generally considered as a congenital pathology, though some consider it as an acquired pathology.,,
Spontaneous closure of DAVF is a rare phenomenon. The majority of case with spontaneous closures, reported so far have been of the cognard type I class. The earliest reported the case of spontaneous closure of type II (a + b) was in 1979 by Endo et al.
Luciani et al. have divided the spontaneously closed DAVF cases into spontaneous and post-traumatic ones; however, they were not able to establish a sound relationship between trauma and spontaneous closure of DAVF. In our case, the patient had a history of a head injury preceding his presentation and diagnosis of DAVF.
The most frequent symptom of DAVF is pulsatile tinnitus followed by a headache., Our patient presented with a headache and vision loss in both the eyes. Some patients have reported a change in symptoms after the closure of DAVF, but in our case, there was no change in headache characteristic of the patient.
Thrombosis of the sinus is considered one of the important factors for the development of DAVF.,, However, thrombosis of sinus and intracerebral hemorrhage causing mass effect and secondary vasospasm have also been considered as mechanisms postulated for spontaneous closure of a fistula.,,,, The disappearance of DAVF can result either from i) direct closure of the shunt or ii) increase in the sinus size due to the structural changes and subsequently leading to the compression of the AV shunts in the wall causing an occlusion of DAVF.,, However, hemorrhage is usually not associated with type I DAVF, which may not explain the spontaneous closure in all cases. Allergic response to the contrast media has also been reported to one of the possible causes of spontaneous closure of DAVF., However, the exact cause of closure of DAVF remains unclear.
To our knowledge, no case has been reported in which a patient presented with increasing headache and worsening vision loss. Our case underwent successful CSF diversion procedure and at follow-up showed resolved papilledema and improvement in vision.
Spontaneous closure of DAVF type II (a + b) is uncommon, whereas it is common in type I DAVF.
We describe a case of type II (a + b) DAVF with spontaneous closure, which is the second case only described in the literature. The mechanism of spontaneous closure of DAVF is still not well understood. This case gives insight to the natural history of type II (a + b) DAVF in which spontaneous closure can also be considered and how a patient can present with complaints of blurring of vision, which should be given timely attention and proper treatment. This case adds to the therapeutic and prognostic implications of type II (a + b) DAVF.
- CSF- cerebrospinal fluid
- CT - computed tomogram
- DAVF- Dural arteriovenous fistula
- DSA - Digital subtraction angiogram
- MRI - magnetic resonance imaging
- MRV- magnetic resonance venogram
- ICP- intracranial pressure.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Informed consent: The patient has consented to the submission of this case report to the journal.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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