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| LETTER TO EDITOR |
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| Year : 2010 | Volume
: 58
| Issue : 1 | Page : 154-155 |
A rare case of metachronous double spinal dural arteriovenous fistula
Amit Dagar1, Manish K Kasliwal1, Ashish Suri1, Subhash Kumar2, Ajay Garg2, Bhawani S Sharma1
1 Department of Neuroradiology, Neurosciences Centre, All India Institute of Medical Sciences, New Delhi - 110 029, India
2 Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi - 110 029, India
| Date of Acceptance | 23-Oct-2009 |
| Date of Web Publication | 8-Mar-2010 |
Correspondence Address:
Bhawani S Sharma
Department of Neuroradiology, Neurosciences Centre, All India Institute of Medical Sciences, New Delhi - 110 029
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0028-3886.60427
How to cite this article:
Dagar A, Kasliwal MK, Suri A, Kumar S, Garg A, Sharma BS. A rare case of metachronous double spinal dural arteriovenous fistula. Neurol India 2010;58:154-5 |
Sir,
Spinal dural arteriovenous fistulas (DAVFs), the most common of spinal vascular malformations consist of an AVF located in the dura mater of the nerve root and/or adjacent spinal dura and is most often, single. [1] Reports of multiple spinal DAVFs are rare; most of which presented synchronously. [2],[3],[4] True metachronous spinal DAVF is a very rare entity. [5]
A 58-year-old male presented with gradual onset spastic paraparesis with bladder symptoms of one year duration. Magnetic resonance imaging (MRI) and spinal angiography revealed the presence of signal changes in the cord with a left T12 DAVF. [Figure 1] Intraoperatively, there was a radicular feeding artery communicating with venous system at the dural sleeve with dilated coronal venous plexus which was coagulated and divided. Patients improved symptomatically gradually over six weeks following surgery and started to walk with support for two years when he again had deterioration in power with worsening of symptoms. Spinal angiogram revealed the presence of a newer DAVF at right L1 level with no evidence of previously treated DAVF at left T12 level. [Figure 2] Previous spinal angiogram were reviewed to rule out preexistence of right L1 DAVF at the time of first spinal angiography, [Figure 2] thus documenting the true development of a metachronous double spinal DAVF. The patient underwent surgery again for the second DAVF with some improvement at a three-month follow-up.
Multiple spinal DAVFs are rare most of them reported were diagnosed either synchronously or were documented during second examination, being missed during the initial angiography. [2],[3],[4] Though the exact pathophysiology of DAVF remains unclear, late onset of symptoms, lack of other associated vascular anomalies, predominant occurrence in males and development of such lesions following trauma and following surgery strongly points towards an acquired etiology. [1],[2],[5] The development of metachronous double DAVF as described by us and Rizvi et al. [5] further adds support to an acquired origin of these vascular spinal lesions.
Various hypotheses have been advocated in cases with multiple DAVF like thrombosis at several sites due to hypercoagulable state or venous stagnation and subsequent thrombosis due to one DAVF. [2],[3],[4],[5] Though the exact mechanism of development of metachronous DAVF as described in the present case remains controversial, chances of venous thrombosis occurring in the spinal/medullary veins or iatrogenic venous injury after surgical treatment of the original spinal DAVF appears to be the most probable etiology. Progressive pial venous thrombosis causing venous ischemia and subsequent spinal cord infarction can cause deterioration a few days after embolization; so can a development of irreversible ischemic damage to the spinal cord. [5],[6] Apart from this, development of a newer DAVF can also lead to failure of treatment with recurrence of symptoms as in the present case. This can also be due to the presence of another synchronous DAVF, which was missed on an initial angiogram as reported by some authors. [4] Though diagnosis of double or multiple metachronous spinal DAVF is rare, its clinical importance cannot be overemphasized as recognition of such cases is essential highlighting the need for careful and thorough performance of spinal angiography at all levels especially during follow up in cases with poor response to treatment.
Magnetic resonance angiography (MRA) can also be a useful non invasive follow-up modality for these patients and can help rule out the development of a metachronous DAVF. The rare occurrence of metachronous double spinal DAVFs as shown in the present case should be considered a possibility accounting for delayed neurological deterioration following successful treatment and a complete spinal angiography at all levels/MRA should be performed to rule out this rare occurrence.
| » References | |  |
| 1. | Rosenblum B, Oldfield EH, Doppman JL, Di Chiro G. Spinal arteriovenous malformations: A comparison of dural arteriovenous fistulas and intradural AVMs in 81 patients. J Neurosurg 1987;67:795-802.  [PUBMED] [FULLTEXT] |
| 2. | Barnwell SL, Halbach VV, Dowd CF, Higashida RT, Hieshima GB, Wilson CB. Multiple dural arteriovenous fistulas of the cranium and spine. Am J Neuroradiol 1991;12:441-5. [PUBMED] |
| 3. | El-Serwi A, Maubon A, Vidal J, Chapot R. Double spinal dural arteriovenous fistulas. Am J Neuroradiol 2006;27:321-3. [PUBMED] [FULLTEXT] |
| 4. | Pierot L, Vlachopoulos T, Attal N, Martin N, Bert S, Chiras J. Double spinal dural arteriovenous fistulas: Report of two cases.Am J Neuroradiol 1993;14:1109-12. [PUBMED] |
| 5. | Rizvi T, Garg A, Mishra NK, Gaikwad SB, Gupta V. Metachronous double spinal dural arteriovenous fistulas.Case report and review of the literature. J Neurosurg spine 2006;4:503-5. [PUBMED] [FULLTEXT] |
| 6. | Larsson EM, Desai P, Hardin CW, Story J, Jinkins JR. Venous infarction of the spinal cord resulting from dural arteriovenous fistula: MR imaging findings.Am J Neuroradiol 1991;12:739-43. [PUBMED] |
[Figure 1], [Figure 2]
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