The Effects of Angiotensin II or Angiotensin 1-7 on Rat Pial Microcirculation during Hypoperfusion and Reperfusion Injury: Role of Redox Stress.

Renin-angiotensin systems produce angiotensin II (Ang II) and angiotensin 1-7 (Ang 1-7), which are able to induce opposite effects on circulation. This study in vivo assessed the effects induced by Ang II or Ang 1-7 on rat pial microcirculation during hypoperfusion-reperfusion, clarifying the mechanisms causing the imbalance between Ang II and Ang 1-7. The fluorescence microscopy was used to quantify the microvascular parameters. Hypoperfusion and reperfusion caused vasoconstriction, disruption of blood-brain barrier, reduction of capillary perfusion and an increase in reactive oxygen species production. Rats treated with Ang II showed exacerbated microvascular damage with stronger vasoconstriction compared to hypoperfused rats, a further increase in leakage, higher decrease in capillary perfusion and marker oxidative stress. Candesartan cilexetil (specific Ang II type 1 receptor (AT1R) antagonist) administration prior to Ang II prevented the effects induced by Ang II, blunting the hypoperfusion-reperfusion injury. Ang 1-7 or ACE2 activator administration, preserved the pial microcirculation from hypoperfusion-reperfusion damage. These effects of Ang 1-7 were blunted by a Mas (Mas oncogene-encoded protein) receptor antagonist, while Ang II type 2 receptor antagonists did not affect Ang 1-7-induced changes. In conclusion, Ang II and Ang 1-7 triggered different mechanisms through AT1R or MAS receptors able to affect cerebral microvascular injury.

De Novo Dural Arteriovenous Fistula on Draining Veins of Previously Treated Pial Arteriovenous Malformation: a Case Report.

A 71-year-old man, with a pial micro-arteriovenous malformation (pAVM) draining into the confluence of the vein of Trolard and the vein of Labbé was surgically removed, sparing these cortical veins. 4-months MR and angiographic controls showed a de novo dural arteriovenous fistula (dAVF) draining into the previously spared cortical veins. It was removed using intraoperative motor evoked potentials (MEP). This is the first case of iatrogenic dAVF developing on the same draining vein of a previously treated pAVM. De novo dAVFs are generally iatrogenic. This case suggests that the unresected venous drainage of an AVM might be the substratum for neo-angiogenetic processes; moreover inflammation related to surgery might be the trigger factor for the development of the dAVF.

Clinical, angiographic, and treatment characteristics of cranial dural arteriovenous fistulas with pial arterial supply.

BACKGROUND: The prevalence of pial arterial supply to cranial dural arteriovenous fistulas (dAVF) and its implication in the management of these fistulas is not well characterized. We performed a retrospective study to characterize pial arterial supply to dural arteriovenous fistulas and the implications for treatment. METHODS: Consecutive patients evaluated over a 12-year period were retrospectively reviewed. Angiograms were reviewed to characterize dAVF angioarchitecture and the presence of pial artery supply. Pial artery supply was categorized as dilated pre-existing dural branches and pure pial supply. We then studied the association between pial artery supply and clinical, angiographic, and treatment features. RESULTS: A total of 201 patients were included of which 27 (13.4%) had pial artery supply. Of these, 11 had supply from dilated pre-existing dural branches, nine had pure pial supply,and seven had both. There was a higher rate of dAVF rupture in the pial supply group (30.8% vs 9.8%, P=0.003) and these fistulas had a higher rate of Borden 2 and 3 (88.9% vs 38.4%, P<0.0001). Fistulas with pial artery supply had similar rates of endovascular and gamma knife treatment, but were more likely to undergo surgery than those without pial supply (25.9% vs 10.4%, P=0.03). Major complication rates were similar between groups (0% vs 1.1%, P=0.55). CONCLUSIONS: More than 10% of dAVFs also have pial supply but this is not a contraindication to embolization. In our study pure pial supply was associated with a more aggressive fistula and was most common in tentorial dAVFs.

Pial arteriovenous fistula with appearance of hemorrhagic tumor: A case report.

INTRODUCTION: Pial arteriovenous fistula (PAVF) is a rare intracranial vascular disease, and its presentation with a huge tumor-resembling thrombus is rarer. PATIENT CONCERNS: A 38-year-old female patient presented with a sudden left-side motor disorder and loss of consciousness. The patient was otherwise in good health and had no history of hypertension or diabetes. During the physical examination, she appeared lethargic and manifested left limb paralysis with level zero muscle strength and a positive pathological reflex. DIAGNOSES: Because imaging failed to rule out a tumor stroke, an intracranial lesion resection was performed immediately. Because the lesion was considered to be a vascular structure, digital subtraction angiography was undertaken before the surgery, and PAVF was diagnosed. INTERVENTIONS: Endovascular embolization was conducted, followed by PAVF and hematoma resection. OUTCOMES: At the 3-month follow up, her left limb muscle strength was level 4, and she could live on her own (Modified Rankin Scale score = 2). CONCLUSIONS: It is noteworthy that PAVF with a large thrombus may appear as a tumor in the initial diagnosis, and therefore it is necessary to perform an intracranial vascular examination in patients with tumor stroke symptoms.

Cerebral amyloid-ß-related angiitis without cerebral microbleeds in a patient with subarachnoid hemorrhage.

Amyloid-ß-related angiitis (ABRA), a subtype of cerebral amyloid angiopathy (CAA), is vasculitis occurring in relation to amyloid-ß (Aß) deposition in the walls of intracranial blood vessels. ABRA is presumed to be caused by some immune response to the deposited Aß. An 81-year-old man on oral anticoagulant therapy complained of headache, nausea, and difficulty with standing after a head injury. Head computed tomography revealed subcortical bleeding in the right temporoparietal lobe, and 3 days after admission, magnetic resonance imaging (MRI) showed subarachnoid hemorrhage (SAH) around the hematoma. Cerebral microbleeds, a characteristic of CAA, were not detected on MRI. On worsening of his symptoms, intracranial brain biopsy and hematoma removal were performed. Intraoperative rapid diagnosis with a frozen section suspected vasculitis, which enabled the prompt initiation of steroid therapy. He was pathologically diagnosed with ABRA (granulomatous angiitis) using a formalin-fixed paraffin-embedded section. Vasculitis was prominent around blood vessels in the pia matter covering the cerebrum. In this case, the inflammatory cells seemed to appear via the subarachnoid space following cerebral hemorrhage and SAH. ABRA seemed to be developed by intracranial hemorrhage in this case.

Clipping of a Pediatric Pial Arteriovenous Fistula Located at Basilar Artery Tip Using a Hybrid Trapping-Evacuation Technique.

BACKGROUND: Intracranial pial arteriovenous fistulas (PAVFs) are rare cerebrovascular lesions with high mortality rates. We report a rare case of pediatric PAVF at the basilar artery tip and its treatment with surgical clipping aided by a trapping-evacuation technique in a hybrid operating room. CASE DESCRIPTION: An 18-month-old boy was admitted with hypoevolutism and 4-month history of weakness in the left extremities. Magnetic resonance imaging showed a giant aneurysm-like malformation in the area of midbrain and pons. Angiography showed a high-flow PAVF fed by the basilar artery and bilateral P1 segments of the posterior cerebral artery, with deep draining veins into the transverse sinus and straight sinus. Given the intrinsic characteristics of the lesion, such as deep location, giant fistula and varix, and multiple feeding arteries, clipping of PAVF was performed in a hybrid operating room aided by a trapping-evacuation technique to clearly identify and block the shunting point. CONCLUSIONS: The successful obliteration of the lesion is reported. In addition, a brief review of literature comparing endovascular embolization, surgical disconnection, and hybrid technique for treatment of PAVF is included.

Salvinorin A preserves cerebral pial artery autoregulation after forebrain ischemia via the PI3K/AKT/cGMP pathway.

This study aimed to investigate the protective effect of salvinorin A on the cerebral pial artery after forebrain ischemia and explore related mechanisms. Thirty Sprague-Dawley rats received forebrain ischemia for 10 min. The dilation responses of the cerebral pial artery to hypercapnia and hypotension were assessed in rats before and 1 h after ischemia. The ischemia reperfusion (IR) control group received DMSO (1 µL/kg) immediately after ischemia. Two different doses of salvinorin A (10 and 20 µg/kg) were administered following the onset of reperfusion. The 5th, 6th, and 7th groups received salvinorin A (20 µg/kg) and LY294002 (10 µM), L-NAME (10 µM), or norbinaltorphimine (norBIN, 1 µM) after ischemia. The levels of cGMP in the cerebrospinal fluid (CSF) were also measured. The phosphorylation of AKT (p-AKT) was measured in the cerebral cortex by western blot at 24 h post-ischemia. Cell necrosis and apoptosis were examined by hematoxylin-eosin staining (HE) and TUNEL staining, respectively. The motor function of the rats was evaluated at 1, 2, and 5 days post-ischemia. The dilation responses of the cerebral pial artery were significantly impaired after ischemia and were preserved by salvinorin A treatment. In addition, salvinorin A significantly increased the levels of cGMP and p-AKT, suppressed cell necrosis and apoptosis of the cerebral cortex and improved the motor function of the rats. These effects were abolished by LY294002, L-NAME, and norBIN. Salvinorin A preserved cerebral pial artery autoregulation in response to hypercapnia and hypotension via the PI3K/AKT/cGMP pathway.

Pial Arteriovenous Fistula: A Brief Review and Report of 14 Surgically Treated Cases.

OBJECTIVE: The authors report their successful experience of treating 14 cases of pial arteriovenous fistula (PAVF) by direct surgery. METHODS: During the period January 2010 to April 2017, 14 patients with PAVF were treated by surgery. Only those patients were selected who had a single arterial feeding channel. There were 9 male patients and 5 female patients, and their ages ranged from 5 to 53 years (average, 19 years). Ten patients were younger than 20 years of age. Five patients presented clinical and radiologic features that suggested hemorrhage from the PAVF. Ten patients had seizures. Two patients had hemispheric symptoms or neurologic deficits at the time of presentation. In 12 patients, there were no gross neurologic deficits. The diagnosis was made on the basis of digital subtraction angiography in all patients and computed tomographic angiography in 8 patients. Angiography revealed that the PAVFs in 8 patients were supplied by the middle cerebral artery, in 5 patients by the anterior cerebral artery, and in 1 patient by branches of the basilar artery. Surgical procedures involved identification of the site of fistula, obliteration of the feeding artery, and resection of the entire venous varix. RESULTS: The PAVF was successfully excluded from circulation in all patients. There were no neurologic deficits related to the surgical procedure. CONCLUSIONS: Direct surgical resection of the entire PAVF is a safe, effective, and probably curative method of treatment.

Surgical Revascularization for Children with Moyamoya Disease: A New Modification to the Pial Synangiosis.

OBJECTIVE: To summarize therapeutic efficacy of modified pial synangiosis in children with moyamoya disease and our experience with this method. METHODS: A retrospective study was conducted to analyze clinical efficacy of modified pial synangiosis in children with moyamoya disease who were treated between October 2002 and August 2015 at our center. Clinical characteristics of these rare cases were summarized, and surgical efficacy was assessed based on long-term follow-up results. RESULTS: We employed modified pial synangiosis to treat 10 children with moyamoya disease; 18 modified pial synangiosis procedures were performed. The study included 2 boys and 8 girls (mean age at disease onset, 6.5 years ± 2.6). According to preoperative digital subtraction angiography, Suzuki grade III was noted in 80% (16/20) of hemispheres, and Suzuki grade II was noted in the remaining hemispheres (4/20). Mean follow-up period was 63.4 months ± 36.0. During the follow-up period, 2 cases of transient ischemic attack were reported. The remaining patients had no evidence of cerebral ischemia, seizures, or cerebral hemorrhage. Postoperative assessments based on Matsushima classification scores showed that patients with grade A revascularization accounted for 66.7% (12/18) of treated hemispheres, patients with grade B accounted for 27.8% (5/18), and patients with grade C accounted for 5.6% (1/18). CONCLUSIONS: Our clinical findings provide data on efficacy and safety of modified pial synangiosis, but analysis of more cases is necessary to draw solid conclusions. A randomized controlled study is required to verify improved surgical efficacy of modified pial synangiosis.

[Effects of vascular peptide bioregulator on cerebral microcirculation of old hypertensive rats].

Using a TV device to study brain microcirculation, we found that after a course of vascular peptide bioregulator the density of microvascular network of pia matter of old hypertensive rats (12 months) sensomotor cortex increased about 1,7 times compared to intact old rates SHR. This perfusion in the tissue of the cerebral cortex and the degree of blood oxygen saturation in the microvasculature of this tissue region raised.

Transfusion of Polynitroxylated Pegylated Hemoglobin Stabilizes Pial Arterial Dilation and Decreases Infarct Volume After Transient Middle Cerebral Artery Occlusion.

BACKGROUND: Polynitroxylation of hemoglobin confers superoxide dismutase-mimetic and peroxidase activity and may protect from reperfusion injury in addition to facilitating oxygen transport. We determined whether transfusion of polynitroxylated PEGylated hemoglobin (PNPH) is protective in the rat filament model of 2 hours of middle cerebral artery occlusion (MCAO). METHODS AND RESULTS: Transfusion of 10 mL/kg of PNPH at 20 minutes of MCAO reduced infarct volume by over 70% (n=10). To determine whether PNPH might act by promoting vasodilation, pial arteriolar diameter in the distal MCA border region was measured in closed cranial windows. With no transfusion, MCAO induced an initial dilation (36±2% ±SE) that subsided by 2 hours (5±4%; n=8). With PNPH transfusion at 20 minutes of MCAO, the initial dilation (31±3%) was better maintained at 2 hours (21±4%; n=7; P<0.02). Delaying PNPH transfusion until 90 minutes of MCAO increased perfusion in the border region from 48±6% of the preischemic baseline to 67±8% (n=8; P<0.005). The effect of PNPH transfusion after reperfusion was also tested. Compared with the control median hemispheric infarct volume of 22% (13% to 34% interquartiles; n=15), infarct volume was reduced to 7% (3% to 13%; n=14 P<0.05) when PNPH was transfused at 4 hours after MCAO (2 hours of reperfusion) but not significantly when transfused at 6 hours (8%; 3% to 35%; n=14) or at 8 hours (12%; 10% to 25%; n=14) after MCAO. CONCLUSIONS: PNPH transfusion has a significant therapeutic window for protection during and after transient MCAO and may act, in part, by stabilizing vascular function and improving collateral blood flow.

Endovascular occlusion of pial arteriovenous macrofistulae, using pCANvas1 and adenosine-induced asystole to control nBCA injection.

Background In large-caliber pial macrofistulae (pMF), the combination of high blood flow velocity and large efferent artery diameter makes control over the endovascular vessel occlusion difficult and may result in the inadvertent venous passage of occlusive devices or embolic agents. Case descriptions Patient 1: A 27-year-old man presented with headache and ataxia. An infratentorial pMF supplied by both superior cerebellar arteries with venous ectasia was found. The first treatment attempt using balloons and coils failed since the position of either device could not be controlled because of a distal diameter of the feeding artery of 8 mm. In a second session a pCANvas1 (phenox) was deployed at the level of the arteriovenous connection and adenosine-induced asystole allowed the controlled injection of nBCA/Lipiodol with partial occlusion of the pMF. A remaining arteriovenous shunt was occluded under asystole in a third session. The procedures were well tolerated, the patient returned to normal and DSA confirmed the occlusion of the fistula. Patient 2: A 13-year-old boy with hereditary hemorrhagic teleangiectasia presented with an intracerebral hemorrhage from an aneurysm of the left MCA. Twelve weeks after the aneurysm treatment a feeding MCA branch (diameter 4.5 mm) of a right frontal pMF was catheterized. The macrofistula was occluded by deployment of a pCANvas1, followed by the injection of nBCAl/Lipiodol under adenosine-induced asystole. Conclusion pCANvas1 and adenosine-induced asystole allow a controlled injection of nBCA/Lipiodol for the endovascular occlusion of high-flow pMF without venous passage of the embolic agent.

Prenatal Diagnosis and Multimodal Neonatal Treatment of a Rare Pial Arteriovenous Fistula: Case Report and Review of the Literature.

BACKGROUND: Intracranial pial arteriovenous fistulas (PAVFs) are direct communications between the arterial and venous system of the brain, with the characteristic absence of a plexiform nidus, as seen in the classic cerebral arteriovenous malformations. These vascular malformations, usually occurring in the pediatric population, very rarely are diagnosed in utero, because of a lack of understanding of the condition and because they may be hard to visualize. CASE DESCRIPTION: We report a rare case of a mass-effect PAVF diagnosed with fetal magnetic resonance imaging, involving the right cerebral hemisphere, fed by a pericallosal artery and associated with a giant venous dilatation. The PAVF initially was managed by the endovascular embolization. The recruitment of a middle cerebral artery feeder and the rapidly enlarging size of the venous pouch with mass effect required subsequent surgery. CONCLUSIONS: The 2-stage multimodal treatment resulted in complete disappearance of the PAVF without complications.

Pial Arteriovenous Fistula and Capillary Malformation-Arteriovenous Malformation Associated with RASA1 Mutation: 2 Pediatric Cases with Successful Surgical Management.

We present case reports of 2 pediatric patients who were both found to have pial arteriovenous fistulas (AVFs) with subsequent genetic analysis revealing mutations in the RASA1 gene. Considering their family history of distinct cutaneous lesions, these mutations were likely inherited as opposed to de novo mutations. Patient 1 had large capillary malformations on the left side of the face and neck, associated with macrocephaly, and presented at the age of 32 months with speech delay, right-sided weakness, and focal seizures involving the right side of the body. Patient 2 presented with proptosis at the age of 9 months, but was otherwise neurologically intact. Given the chance for definitive single-stage control of vascular shunt (obviating chances for radiation exposure with endovascular treatment) and surgically accessible location of these intracranial lesions, both patients were treated with surgery with excellent clinical and radiological outcome. In general, given the high mortality secondary to severe congestive heart failure when treated conservatively, the goal of treatment in cortical AVF in young children, even when asymptomatic, is rapid control of the shunt. This was achieved successfully in our cases - both patients experienced significant symptomatic improvement following surgery and remained neurologically stable in the subsequent follow-up visits.

Modelling of subarachnoid space width changes in apnoea resulting as a function of blood flow parameters.

During apnoea, the pial artery is subjected to two opposite physiological processes: vasoconstriction due to elevated blood pressure and vasorelaxation driven by rising pH in the brain parenchyma. We hypothesized that the pial artery response to apnoea may vary, depending on which process dominate. Apnoea experiments were performed in a group of 19 healthy, non-smoking volunteers (9 men and 10 women). The following parameters were obtained for further analysis: blood pressure, the cardiac (from 0.5 to 5.0Hz) and slow (<0.5Hz) components of subarachnoid space width, heart rate, mean cerebral blood flow velocity in the internal carotid artery, pulsatility and resistivity index, internal carotid artery diameter, blood oxygen saturation and end-tidal carbon dioxide. The experiment consisted of three apnoeas, sequentially: 30s, 60s and maximal apnoea. The breath-hold was separated for 5minute rest. The control process is sophisticated, involving internal cross-couplings and cross-dependences. The aim of work was to find a mathematical dependence between data. Unexpectedly, the modelling revealed two different reactions, on the same experimental procedure. As a consequence, there are two subsets of cardiac subarachnoid space width responses to breath-hold in humans. A positive cardiac subarachnoid space width change to apnoea depends on changes in heart rate and cerebral blood flow velocity. A negative cardiac subarachnoid space width change to apnoea is driven by heart rate, mean arterial pressure and pulsatility index changes. The described above two different reactions to experimental breath-hold provides new insights into our understanding of the complex mechanisms governing the adaptation to apnoea in humans. We proposed a mathematical methodology that can be used in further clinical research.

Rete mirabile associated with pial arteriovenous fistula: imaging features with literature review.

A rete mirabile is a vascular network of intercommunicating small arteries or arterioles that replace the definitive adult artery supplying the brain. It supplies the brain in lower mammals but is not seen in normal human embryological development. A 26-year-old man presented with worsening tinnitus that was interfering with his sleep. On CT and digital subtraction angiography he was found to have a temporal lobe pial arteriovenous fistula with bilateral carotid and vertebral rete mirabile. The patient was offered open surgical and endovascular treatment options for pial arteriovenous fistula but he refused both and opted for conservative medical management. At 6-month follow-up he continued to have pulsatile tinnitus but was otherwise neurologically normal. We present the first described association of rete mirabile with pial arteriovenous fistula and discuss its clinical presentation and imaging features, with a review of the literature for bilateral carotid and vertebral rete mirabile.

Tetrahydrobiopterin rescues impaired responses of cerebral resistance arterioles during type 1 diabetes.

Our goal was to test the hypothesis that administration of tetrahydrobiopterin (BH4) would improve impaired endothelial nitric oxide synthase-dependent dilation of cerebral arterioles during type 1 diabetes. In addition, we examined the influence of BH4 on levels of superoxide in brain tissue. In vivo diameter of cerebral arterioles in nondiabetic and diabetic rats was measured in response to endothelial nitric oxide synthase-dependent agonists (acetylcholine and adenosine 5'-diphosphate) and an endothelial nitric oxide synthase-independent agonist (nitroglycerine) before and during application of BH4 (1.0 µM). We also measured levels of superoxide from cortex tissue in nondiabetic and diabetic rats under basal states and during BH4 Acetylcholine and adenosine 5'-diphosphate dilated cerebral arterioles in nondiabetic rats, but this vasodilation was significantly impaired in diabetic rats. In contrast, nitroglycerine produced similar vasodilation in nondiabetic and diabetic rats. Application of BH4 did not enhance vasodilation in nondiabetic rats but improved impaired cerebral vasodilation in diabetic rats. Basal superoxide levels were increased in cortex tissue from diabetic rats, and BH4 reduced these levels to that found in nondiabetic rats. Thus, BH4 is an important mediator of endothelial nitric oxide synthase-dependent responses of cerebral arterioles in diabetes and may have therapeutic potential for the treatment of cerebral vascular disease.

Surgical Outcome for Moyamoya Disease: Clinical and Perfusion Computed Tomography Correlation.

OBJECTIVES: To compare surgical outcome both radiologically and clinically after interventions for patients with Moyamoya disease. METHODS: This retrospective observational study included 25 patients who were treated surgically for Moyamoya disease in the past 14 years. Clinical outcomes were analyzed by subgroups stratified by age, disease presentation, and surgical intervention. Serial postoperative brain computed tomography perfusion records were analyzed with respect to the cerebral blood flow and cerebrovascular reserve capacity (CVRC) of the middle cerebral artery territory. Changes in both the intervention (n = 23) and nonintervtion (n = 9) cerebral hemispheres were compared. RESULTS: All children treated by synangiosis (n = 9), all adults receiving synangiosis (n = 5), and 88.9% of adults undergoing bypass (n = 9) had no neurologic deterioration, with a duration of at least 50.6 months, 85.7 months, and 27.7 months, respectively. Radiologically, CVRC improved more markedly after bypass surgery than synangiosis, particularly 12-24 months postoperatively (51.1% vs. -2.86%). The hemispheres that did not undergo intervention showed similar improvement in cerebral blood flow over time compared with the hemispheres that did undergo intervention, after surgery was performed. CONCLUSIONS: Bypass surgery improved CVRC greater than synangiosis, which may correlate with decreased future stroke risks. The decision for bypass is to be balanced with a greater risk of postoperative neurologic deterioration in adults after this procedure. The hemisphere that did not undergo intervention also appeared to benefit from surgery performed on the contralateral brain.

The Role of HMGB1 in Pial Arteriole Dilating Reactivity following Subarachnoid Hemorrhage in Rats.

High-mobility group box 1 protein (HMGB1) has been implicated in inflammatory responses, and is also associated with cerebral vasospasm after subarachnoid hemorrhage (SAH). However, there are no direct evident links between HMGB1 and cerebral vasospasm. We therefore investigated the effects of HMGB1 on pial arteriole reactivity following SAH in rats. We initially found that SAH induced a significant decrease in pial arteriole dilating responses to sciatic nerve stimulation (SNS), hypercapnia (CO2), and the topical suffusion of acetylcholine (ACh), adenosine (ADO), and s-nitroso-N-acetylpenicillamine (SNAP) over a 7-day period after SAH. The percent change of arteriolar diameter was decreased to the lowest point at 48 h after SAH, in response to dilating stimuli (i.e., it decreased from 41.0 ± 19.0% in the sham group to 11.00 ± 0.70% after SNS) (n = 5, p < 0.01). HMGB1 infusion in the lateral ventricle in normal rats for 48 h did not change the pial arteriole dilating response. In addition, inhibitors of HMGB1-receptor for advanced glycation end-product or HMGB1-toll-like receptor 2/4 interaction, or the HMBG1 antagonist did not improve pial arteriole reactivity 48 h after SAH. These findings suggest that HMGB1 may not be a major player in cerebral vascular dilating dysfunction after SAH.

Multiple Dural and Pial Arteriovenous Fistulae in a Twenty-Four-Year-Old Woman in the Setting of Superior Sagittal Sinus Thrombosis: Case Report and Review of Literature.

CASE: A 24-year-old woman presented with headache, nausea, and vomiting, and was found to have chronic superior sagittal sinus (SSS) thrombosis and multiple dural arteriovenous fistulae (dAVFs). Despite anticoagulant therapy and successful recanalization of her sinus, her fistulae persisted, and she developed additional separate pial arteriovenous fistulae (pAVFs). Her fistulae were treated with staged endovascular embolization, open clipping, and gamma knife radiosurgery over the course of 10 months. Complete resolution of SSS thrombosis and all arteriovenous fistulae (AVFs) was noted on cerebral angiogram performed 18 months from initial presentation. DISCUSSION: dAVFs have frequently been associated with venous sinus thrombosis. Sinus thrombosis resulting after endovascular or surgical treatment of dural arteriovenous fistulous connections has been reported in literature and is considered a possible complication of treatment. Multiple dAVFs and pAVFs are rare and often require multimodal staged approaches for definitive treatment. CONCLUSION: We report a case of chronic sagittal sinus thrombosis resulting in multiple AVFs requiring staged multimodal treatment with successful resolution of the fistulous connections. Furthermore, upon reviewing the literature addressing multiple dAVFs and the treatment of such lesions using endovascular, microsurgical, and stereotactic radiosurgery techniques, we elucidate the success a multimodal approach to therapy can afford for the unique challenges associated with multiple lesions.