Sinus reconstruction therapy for superior sagittal sinus dural arteriovenous fistula caused by parasagittal meningioma invasion: a case report

Abstract

A dural arteriovenous fistula (dAVF) involving the superior sagittal sinus (SSS) is relatively rare, and its clinical course is usually aggressive. Its concomitance with a tumor has rarely been reported. Here, we present a case of SSS dAVF due to meningioma invasion, which was treated with sinus reconstruction and endovascular embolization. A 75-year-old man who had undergone tumor resection for parasagittal meningioma 4 years prior presented with intra-ventricular hemorrhage. Computed tomography angiography and magnetic resonance imaging revealed recurrent tumor invasion into the SSS causing occlusion. Cerebral angiography revealed multiple shunts along the occluded segment of the SSS, diffuse deep venous congestion, and cortical reflux. Borden type 3 SSS dAVF was diagnosed. We first performed direct tumor resection, followed by stenting for the occluded SSS and partial embolization of the shunts. After a 6-month interval, transvenous occlusion of the SSS was performed along the stent, resulting in complete obliteration of the dAVF. Sinus reconstruction therapy was effective in the immediate improvement of venous hypertension, obtaining the access route to the fistulas, and eradicating the shunts.

Introduction

Dural arteriovenous fistulas (dAVFs) are abnormal arteriovenous shunts within the dura. Although the pathogenesis of dAVFs is unknown, they are considered acquired lesions secondary to various conditions, including infection, trauma, surgery, and thrombophlebitis.^1–3 DAVF of the superior sagittal sinus (SSS) is a relatively rare type and accounts for 5–8% of all dAVFs.^4,5 SSS is the main drainage route from the bilateral cerebral cortices; therefore, dAVF involving the SSS can easily cause elevated intracranial pressure, resulting in fatal complications, such as intracranial hemorrhage, venous infarction, and epilepsy. ⁶ Although symptomatic SSS dAVF needs urgent management to prevent undesirable clinical outcomes, the treatment strategy is still controversial and often demands multimodal therapy.^7,8

DAVFs sometimes occur at the venous sinus invaded by meningioma or distant from the site, and the possibility of hyperexpression of angiogenic substances or venous sinus hypertension by meningioma is reported.^9,10 Most previous cases involved the transverse sinus (TS) or sigmoid sinus (SS), and the therapeutic strategy for the dAVF involving SSS caused by sinus occlusion due to parasagittal meningioma is not well documented.^3,10–15 This rare and complicated condition makes treatment for SSS dAVF challenging. Here, we report a case of SSS dAVF caused by SSS occlusion due to meningioma invasion and present the technical details of the staged therapy of sinus reconstruction and endovascular embolization.

Case presentation

A 75-year-old man with symptoms of strange behavior and difficulty communicating was transferred to our hospital. Four years prior, he had undergone removal of a parasagittal meningioma (transitional meningioma, World Organization grade (1) that had invaded SSS, and surgery was performed with a part of the tumor remaining (Figures 1(a) and (b)). Physical examination on arrival revealed disturbance of consciousness and apraxia. Computed tomography revealed intra-ventricular hemorrhage. Magnetic resonance imaging showed diffuse deep venous ectasia and slight enlargement of the residual tumor, which measured 1×3 cm and was invading the SSS (Figures 1(c) and (d)). Cerebral angiography showed an SSS dAVF with multiple shunts supplied by the bilateral posterior convexity branches of the middle meningeal artery, superficial temporal arteries (STAs), and occipital arteries (OAs) (Figures 2(a), (b), and (c)) reflux, drainage toward the internal jugular vein via the cavernous sinus and the Galenic system, and occlusion of the posterior third of the SSS (Figures 2(d) and (e)). Borden type 3 SSS dAVF with sinus occlusion due to meningioma invasion was diagnosed (Figure 2(f)).

Figure 1.

Axial (a) and sagittal (b) views of contrast-enhanced T1-weighted MRI at the first treatment show the parasagittal meningioma of size 27 × 26 × 28 mm in the left occipitalregion infiltrating the posterior third of the SSS. Axial (c) and sagittal (d) views of contrast-enhanced T1-weighted MRI at admission show the remnant parasagittal meningioma infiltrating the posterior third of the SSS. The presence of multiple enlarged vessels along the leptomeningeal space suggests venous hypertension, probably related to a dural arteriovenous fistula. MRI, magnetic resonance image; SSS, superior sagittal sinus..

Figure 2.

Lateral view of right external carotid artery angiography showing the multiple shunts in the two regions of the SSS, one of which is supplied from the middle meningeal artery and superficial temporal artery (a), and the other is supplied from the occipital artery (b). Lateral view of right CCA angiography at the arterial phase (c) shows the cortical venous refluxes (black arrows) and the occluded posterior third of the SSS (white arrowheads). Anteroposterior (d) and lateral (e) views of right CCA angiography at the venous phase show diffuse venous congestion and the occluded posterior third of the SSS (white arrowheads). Schematic view of the positional relation between the tumor and two shunt points showing one of the fistulous points was just above the tumor, and the other was distal to it (f). SSS, superior sagittal sinus; CCA, common carotid artery.

We performed staged treatment involving sinus angioplasty and shunt embolization. First, we resected the tumor inside the SSS to facilitate catheterization to access the lesion. We approached the tumor using the previous skin incision and parieto-occipital craniotomy. After opening the dura matter, the tumor was resected until bleeding was observed at the edges (Figures 3(a) and (b)). Finally, the sinus was tightly closed. Angioplasty using balloon-expandable stents was performed 3 weeks later. We selected a guidewire with a high tip load to penetrate the occluded SSS. After passing through the lesion with a 0.035-inch guidewire (Radifocus; Terumo, Tokyo, Japan), a 0.018-inch hard-tip guidewire (Treasure 12; Tip load 12.0 gf, ASHI INTECC, Aichi, Japan) and microcatheter (Excelsior SL-10; Stryker Neurovascular, Fremont, California, USA) were passed through the occluded segment (Figure 4(a)). The diameters of the normal venous sinus proximal and distal to the occluded segment were 4.2 and 3.2 mm, respectively. Then, a 3.5×20 mm balloon catheter (Gateway; Stryker) was used to perform percutaneous transluminal angioplasty, and three Multilink Duet coronary stents (Guidant Corp., Indianapolis, Indianapolis, USA) were placed (Figure 4(d)). The first stent (3×40 mm) was deployed from the distal side of the occluded lesion, and the second stent (4×40 mm) was partially overlapped and positioned to avoid occlusion of the orifice of the torcular herophili. The third stent (3.5×18 mm) was placed just distal to the occluded lesion where the sinus was stenosed and multiple shunt channels flowed in. After deployment of each stent, balloon inflation was performed to fix the stent in the sinus wall. We then attempted trans-arterial and transvenous embolization for the dAVF using Onyx-18 (Medtronic, Minneapolis, Minnesota, USA) (Figures 4(b) and (e)). A microcatheter was transvenously navigated through the patent SSS and placed in the shunted venous pouch. Onyx-18 was injected under sinus protection with a balloon catheter. It migrated from the fistulous channels into the arterial feeders, resulting in partial embolization of the shunts. We then performed embolization from the left STA and right OA using a Magic 1.2FM (Balt, Montmorency, France) flow-guide catheter. Injection of Onyx-18 infiltrated multiple shunts along the venous pouch. Post-treatment angiography revealed patency of the SSS, improved venous congestion, and decreased cortical reflux (Figures 4(c) and (f)). The dAVF was converted from Borden type 3 to type 2. There was marked improvement in the patient’s intellectual function, and he was discharged from the hospital with a modified Rankin Scale score of 1.

Figure 3.

The intraoperative view shows the meningioma (dotted line) infiltrating the superior sagittal sinus (a). The tumor was resected until the sinus wall could be seen (b).

Figure 4.

Anteroposterior view of the roadmap image (a) showing the penetration of the occluded lesions by a 0.035-inch guidewire (long arrow) followed by a micro-guide wire (short arrow). Lateral fluorography (d) shows the deployment of the balloon-expandable stents at the superior sagittal sinus (short arrows: the first stent, long arrows: the second stent). LAO (b) and lateral (e) fluorography showing the third stent (black arrows), Onyx casts injected from the right superficial temporal artery (black arrowheads), and right occipital artery (white arrowheads). Post-treatment LAO (c) and lateral (f) view of the right CCA angiography show the patent SSS and reduced venous congestion. LAO, left anterior oblique; CCA, common carotid artery; SSS, superior sagittal sinus.

At the 6-month follow-up angiography, occlusion of the SSS by venous thrombosis and an exacerbation of the dAVF was observed. We again passed through the occluded lesion and placed the microcatheter in the SSS distal to the third stent. The posterior part of the SSS was occluded from the fistulous points to the proximal side of the second stent with seven coils and Onyx (Figures 5(a) and (d)). Post-treatment angiography confirmed complete disappearance of the dAVF (Figure 5(b), (c), (e), and (f)). No neurological abnormalities or complications were observed after the treatment. At the 2-year follow-up, angiography showed no recurrence of the shunts, and the patient had no neurological deficit.

Figure 5.

Anteroposterior view of the fluorography (a) shows the Onyx casts in the superior sagittal sinus, superficial temporal artery, and occipital artery. Sagittal cone-beam computed tomography (d) shows the coil mass and Onyx casts within the stent. Post-treatment anteroposterior and lateral views of the right CCA angiography at the arterial phase (b and e) and venous phase (c and f) show the disappearance of the dAVF and improved venous congestion. CCA, common carotid artery; dAVF, dural arteriovenous fistula.

Discussion

This report presents a rare case of SSS dAVF associated with recurrent parasagittal meningioma that was treated with a combination of direct surgery and endovascular treatment. In addition, we demonstrated the usefulness of sinus reconstruction.

The therapeutic management for dAVFs, including follow-up observation, the surgical approach, radiosurgery, and endovascular treatment, should be chosen based on clinical conditions and the lesion angioarchitecture, with the reference to the grading system of dAVF, such as Borden classification.^1,16 In our case, Borden type 3 dAVF with occluded SSS and leptomeningeal venous drainage caused venous hypertension, resulting in aggressive symptoms. Two previous reports presented SSS dAVFs associated with contiguous parasagittal meningioma, one of which was Borden type 3 with occluded SSS, and the other was Borden type 2 with patent SSS (Table 1). Both presented severe symptoms and were treated by partial embolization of the shunt via a trans-arterial approach and tumor resection with clipping of the draining vein.^17,18 However, in the present case, we expected that it would be difficult to eradicate all shunt points by surgical approach or trans-arterial embolization because of the multiple fistulous points and fine arterial networks. Tumor concomitance made the management of this case even more challenging, and transvenous embolization was not feasible due to the occluded SSS. Therefore, we performed staged therapy to recanalize the SSS and embolize the shunts.

Table 1.

Clinical characteristics of the patients in the reviewed literature.

Author & Published year	Age	Sex	Symptoms	Hemorrhagic presentation	Feeders	Drainers	Sinus occlusion	Borden type	Treatment procedure
Toledo et al. 2010	60	M	Confusion, aphasia	No	Bil. MMA STA	Cortical vein, Galenic system	Yes	III	TAE + tumor resection, surgical shunt dissection
Nakazawa et al. 2022	71	M	Epilepsy	Yes, left frontal subcortical hemorrhage	Bil. MMA, STA, Rt. ACA	SSS	No	II	TAE + tumor resection, surgical shunt dissection
Present case 2022	71	M	Consciousness disturbance and apraxia	Yes, IVH	Bil. MMA, STA, OA	Cortical vein, Galenic system	Yes	III	Tumor resection + TAE, TVE

M: male, F: female, IVH: intra-ventricular hemorrhage, Bil: bilateral, MMA: middle meningeal artery, STA: superficial temporal artery, ACA: anterior cerebral artery, OA: occipital artery, SSS: superior sagittal sinus, TAE: trans-arterial embolization, TVE: transvenous embolization

We initiated treatment by reconstructing the sinus with stent placement after tumor resection, which was useful in this case for three reasons: (1) as rescue therapy for venous congestion, (2) to obtain the access route for the shunt points, and (3) to secure the complete occlusion of the multiple shunt channels. First, sinus reconstruction was performed to restore the antegrade venous flow. Urgent treatment was necessary for the multiple Borden type 3 dAVFs, as in our patient, to reduce the risk of further hemorrhage.^8,19 The possibility of rebleeding due to venous hypertension is also much higher in patients with a previous hemorrhage. ²⁰ We tentatively reconstructed the SSS and downregulated the severity of dAVF by transforming it from Borden type 3 to type 2. The significance of preserving the venous flow of the affected sinus in dAVF has been reported, and sinus stent insertion with balloon angioplasty can lead to favorable functional and imaging outcomes.^21–24 Second, we placed a stent to obtain the access route to the fistulous channels. One of the fistulous points was just above the lesions of tumor invasion, and the other shunt points were distal to it, which limited the access to the fistulas via the internal jugular vein. There was another option of transcranial embolization by direct puncture of SSS; however, this method could not be applied to our patient because of the presence of the tumor.^25,26 Although SSS was unexpectedly reoccluded by the sinus thrombus, the stent maintained the lumen of SSS and enabled the catheter to pass through the occluded segment during the second endovascular treatment. Third, the placed stent covered the diffuse shunt channels and allowed exact obliteration of the lesions along the stent. The stent fixed the sinus lumen by compressing the residual tumor or fibrous tissue against the sinus wall, which enabled tight packing of the coil and Onyx infiltration inside the SSS. Closing the fistulas by the dilated sinus was also an expected effect, according to a previous report. ²⁷

Our treatment has some limitations. It is associated with the risk of sinus thrombosis and is time-consuming. In addition, it may not be feasible in all cases, and careful imaging follow-up is required for the detection of tumor and dAVF recurrence. However, given the complexity and multiplicity of the shunts, this strategy was compatible with the requirements in the present case, where urgent control of venous hypertension and subsequent eradication of the shunts were necessary.

Conclusion

This case report describes a Borden type 3 SSS dAVF caused by meningioma invasion. The findings and outcomes highlight the feasibility and efficacy of sinus reconstruction for the management of such cases. Staged therapy involving sinus angioplasty and trans-arterial/venous embolization could be a useful treatment option for this rare and complex shunt lesion.

ORCID iD

Shogo Shima https://orcid.org/0000-0002-2128-5602