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Intracranial Dural Arteriovenous Fistula with Venous Reflux to the Brainstem and Spinal Cord Mimicking Brainstem Infarction —Case Report—

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Intracranial Dural Arteriovenous Fistula with Venous Reflux to the Brainstem and Spinal Cord Mimicking Brainstem Infarction —Case Report— p024 p.1 [100%] Neurol Med Chir (Tokyo) 44, 24¿28, 2004 Intracranial Dural Arteriovenous Fistula With Venous Reflux to the Brainstem and Spinal Cord Mimicking Brainstem Infarction —Case Report— Jun LI*,#, Masayuki EZURA**,AkiraTAKAHASHI**,andTakashiYOSHIMOTO*** *Department of Neuroendovascular Therapy, Kohnan Hospital, Sendai, Miyagi; Departments of **Neuroendovascular Therapy and ***Neurosurgery, Tohoku University School of Medicine, Sendai, Miyagi; #Department of Neurology, Qingdao Municipal Hospital, Qingdao, P.R.C. Abstract A 73-year-old man presented with a rare transverse sinus dural arteriovenous fistula (dAVF) with venous reflux to the brainstem and medulla manifesting as brainstem and spinal cord edema mimicking brainstem infarction. Complete occlusion of the fistula was achieved by transvenous embolization, resulting in angiographic cure of the fistula and progressive improvement of the symptoms. Intracrani- al dAVFs with perimedullary venous drainage, type V according to the Cognard classification, are rare lesions with distinctive clinical, radiological, and therapeutic aspects. This case demonstrates that the symptoms of dAVF with perimedullary venous reflux are variable, so dAVF should be considered in patients with clinical and radiological findings suggestive of congestion in the brainstem and spinal cord. Dysfunction of the medulla and spinal cord caused by venous hypertension is the most probable cause of the neurological symptoms in such cases. Interventional therapy can lead to angiographic cure and resolution of the symptoms. Key words: dural arteriovenous fistula, vascular myelopathy, venous hypertension, embolization Introduction We present a rare case of transverse sinus dAVF with venous reflux to the brainstem and medulla Dural arteriovenous fistulae (dAVFs) are character- manifesting as brainstem and spinal cord edema ized by abnormal shunting of blood between the mimicking brainstem infarction. arterial system (meningeal branches and rarely cortical branches of the external and internal Case Report carotid and vertebral arteries) and the venous system (dural sinus or close structures), without the A 73-year-old man developed dizziness, nausea, presence of a normal intervening capillary bed. vomiting, and progressive motor weakness in the dAVFs account for 10% to 15% of all intracranial bilateral lower extremities within 3 days. T2-weight- arteriovenous malformations (AVMs).1) Spinal ed magnetic resonance (MR) imaging revealed a dAVF with perimedullary venous drainage is a well- hyperintense area in the pontomedullary region, as known cause of vascular myelopathy. However, well as a hyperintense area in the left temporal lobe, intracranial dAVF with venous drainage into the which was not identified at that time (Fig. 1). He spinal veins is far less common and may be associ- had no significant medical history. One week later, ated with myelopathy. The diagnosis of this uncom- T2-weighted MR imaging showed a hyperintense mon type of drainage is usually challenging because area in the left temporal lobe. He was treated under a the presenting symptoms are variable and often diagnosis of acute cerebral infarction. The patient related to dysfunction of the spine, rather than the was discharged with right leg paresis. Three months brain. later, the patient suddenly developed quadriplegia, unconsciousness, and dyspnea. Brain MR imaging Received April 18, 2003; Accepted August 27, 2003 revealed no new lesion. The diagnosis was recur- 24 p024 p.2 [100%] Type V dAVF Mimicking Brainstem Infarction 25 Fig. 1 Axial fluid-attenuated inversion recovery Fig. 2 Serial sagittal T2-weighted magnetic magnetic resonance images at the initial resonance images in the cervical plane, 7 onset showing hyperintense areas (arrow)in months (A), 8 months (B), and 11 months the medulla oblongata (A) and left temporal after onset (C), and 5 days after emboliza- lobe (B). tion (D), showing changes in the size of the hyperintense area (arrow) with time. Note the obvious shrinkage of the hyperintense lesion and disappearance of the flow void rence of acute cerebral infarction. The patient was ahead and beyond the medulla oblongata 5 intubated and ventilated and given ischemic ther- days after embolization. apy. He gradually regained consciousness. After rehabilitation, he was discharged with bowel and bladder dysfunction and confined to a wheelchair. through a microcatheter (Transit II; Cordis Co., Comparison of the serial MR images revealed that Miami, Fla., U.S.A.) via the retrograde left internal the size and density of lesions in the medulla and jugular vein approach. Postembolization an- temporal lobe changed with time. MR imaging also giography showed angiographic cure of the fistula revealed hyperintensity from the medulla to the level (Fig. 3D, E). of C-5, and a flow void (Fig. 2A–C). AVM was After the interventional therapy, the patient's suspected. He was referred to our hospital one year neurological status progressively improved, and MR after the initial onset. imaging revealed obvious shrinkage of the hyperin- On admission, the patient had clear consciousness tensity 5 days after the therapy (Fig. 2D). and no cranial nerve disturbance. He had normal strength in the upper extremities, but severe Discussion weakness in the proximal lower extremities. The deep-tendon reflexes were normal in both arms and Only a few cases of intracranial dAVF with hyperactive in the legs. Babinski's reflex was present spinal perimedullary venous drainage have been bilaterally. Pinprick sensation was decreased in the reported.2–4,6–8,10,15–17,19–30) The most common symp- S-2 dermatome. Cerebral angiography demonstrated tom is ascending myelopathy, starting as loss of a dAVF in the left transverse sinus, type V according sensationandmotorfunctioninthelowerextremi- to the Cognard classification, fed by the left middle ties and extending to the upper extremities. Sphinc- meningeal artery and meningeal branches of the ter function is impaired. Brainstem symptoms are occipital artery, and drained by the ipsilateral sig- the last to appear and are related to the pattern of moid sinus, jugular vein, and anterior and posterior the venous drainage, which involves the anterior spinal veins (Fig. 3A–C). There was severe stenosis pontomesencephalic vein.8) Review of 37 cases in the left sigmoid sinus. In addition, the meningeal concluded that the symptoms are usually progres- branch from the right ascending pharyngeal artery sive (28 of 37 cases), but acute onset of neurological acted as a feeder across the midline to the left disorders occurs in 25% of cases (8 of 37 cases). transverse sinus. Bulbar dysfunction was reported in seven cases.22) Endovascular embolization was selected as The neurological symptoms of our patient were treatment. Complete occlusion of the fistula was most likely related to spinal cord dysfunction (bowel achieved by transvenous embolization of the trans- and bladder dysfunction and paraparesis) as well as verse sinus using interlocking detachable coils brainstem signs such as unconsciousness and (Boston Scientific, Boston, Mass., U.S.A.) delivered dyspnea. The symptoms of our patient are similar to Neurol Med Chir (Tokyo) 44, January, 2004 p024 p.3 [100%] 26 J. Li et al. Fig. 3 A: Left external carotid angiogram, lateral view, showing two shunts from the middle menin- geal artery to the transverse sinus (arrows), and one shunt from the occipital artery to the transverse sinus (curved arrow). B: Selective angiogram of the left occipital artery, lateral view, showing cortical vein reflux through the vein of Labb áe to the sylvian vein (arrow)and severe stenosis in the left transverse sinus (arrowhead). C: Lateral view of the left occipital artery, late phase, showing anterior and posterior spinal vein reflex (arrows). D: Postopera- tive left external carotid angiogram showing the angiographic cure of the fistula. E: Radio- graph of the brain, lateral view, showing coils filling the transverse sinus (arrow). those of previously reported cases of intracranial symptoms is spinal cord venous hypertension. The dAVFs, in which the patients presented with my- high pressure of retrograde arterial flow into the elopathy first and brainstem symptoms appeared venous systems is thought to be an important cause later. However, the acute onset of the symptoms and of venous ischemia. The severe stenosis of the trans- hyperintensity in the pontomedullary region led to verse sinus was the likely cause of the retrograde an incorrect initial diagnosis. venous drainage into the vein of Labb áe and the Spinal cord dysfunction has several causes14): anterior pontomesencephalic and spinal perimedul- diversion or ``stealing'' of blood from the spinal lary veins. Therefore, a transverse sinus dAVF may cord, cord compression by dilated vascular struc- result in spinal cord venous hypertension, thus tures, hemorrhage, and chronic venous hyperten- behaving exactly like dAVFs that drain directly into sion within the spinal cord. Spinal cord ischemia the spinal veins.9) Ourcaseisagoodexampleofthis caused by arterial steal could be excluded in this phenomenon. case because of the low-flow fistula, and the external Venous congestion in the spinal cord and brain- carotid artery does not supply the spinal cord. No stem of our patient may have been caused by trans- cord compression, hemorrhage, or spontaneous verse sinus AVF reflux to the anterior and posterior thrombosis were detected in this patient. The only spinal veins via well-described anastomotic venous
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