Abstract
Introduction and importance
Bilateral cavernous sinus (CS) dural arteriovenous fistulas (DAVFs) are uncommon, and transarterial embolization (TAE) via the pharyngeal trunk of the ascending pharyngeal artery (AphA) has rarely been reported.
Patient presentation
A 59-year-old female presented with a 1-month history of mild headache. On physical examination, no positive signs were found. Digital subtraction angiography revealed bilateral CS DAVFs. TAE via the pharyngeal trunk of the AphA using the “pressure cooker” technique was performed. After the distal pharyngeal trunk was catheterized with a Marathon microcatheter, the pharyngeal trunk was coiled, and the “pressure cooker” effect was established. The Onyx-18 liquid embolic system was administered, effectively obliterating most of the bilateral CS DAVFs. A small residual left fistula was embolized via the middle meningeal artery (MMA) and accessory meningeal artery (AMA). Postoperatively, the patient suffered an asymptomatic infarction of the head of the caudate nucleus due to Onyx migration into the intracranial artery and minor paralysis of the left side of the face due to injury to the cranial nerve branches of the MMA and AMA. One month later, the patient appeared to have recovered well.
Clinical discussion
For bilateral CS DAVFs, TAE via the pharyngeal trunk of the AphA may be selected when the transvenous route is occluded and when the AphA route is easily accessible. The “pressure cooker” technique was helpful.
Conclusion
In select cases, bilateral CS DAVFs can be embolized via the pharyngeal branch of the AphA. However, possible stroke from dangerous anastomoses must be considered.
Keywords: Cavernous sinus, Bilateral dural arteriovenous fistulas, Ascending pharyngeal artery, Embolization, “Pressure cooker” technique
Highlights
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Bilateral CS DAVFs are uncommon.
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Trans-AphA has rarely been reported.
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The “pressure cooker” technique is helpful.
Abbreviations
- AMA
accessory meningeal artery
- AphA
ascending pharyngeal artery
- CS
cavernous sinus
- DAVF
dural arteriovenous fistula
- DSA
digital subtraction angiography
- EVT
endovascular treatment
- MMA
middle meningeal artery
- TAE
transarterial embolization
1. Introduction
Bilateral cavernous sinus (CS) dural arteriovenous fistulas (DAVFs) are defined as fistulas involving the bilateral CS, with each fistula being fed by one or both carotid arteries [1]. They are rare. Jong et al. reported that bilateral CS DAVFs represent 7.7 % of all cases of CS DAVF [2]. Fay et al. reported that the rate of bilateral CS DAVFs was 14.2 % [3]. When patients with bilateral CS DAVFs present with severe neuroophthalmologic deficits or are at risk of intracranial hemorrhage, aggressive treatment is necessary. Currently, transvenous embolization (TVE) is still the first treatment choice for bilateral CS DAVFs. However, when the TVE route is occluded and there is an easily accessible pharyngeal trunk of the ascending pharyngeal artery (AphA) route, transarterial embolization (TAE) via the AphA can be used to treat bilateral CS DAVFs, which has rarely been reported. Thus, we report such a case.
Our work is in line with the SCARE criteria, which address the importance of patient privacy, consent, ethical considerations, and the need for clear and concise reporting of surgical procedures and outcomes [4].
2. Case presentation
A 59-year-old female presented with a 1-month history of mild headache. She was previously healthy and had no history of systemic hypertension or diabetes mellitus. On physical examination, there were no positive findings. She had grade V muscle strength in her limbs. Magnetic resonance (MR) imaging revealed enhancement of the CS region (Fig. 1A). MR angiography revealed abnormalities in the vessels of the CS region (Fig. 1B). No acute infarction was detected on MR images (Fig. 1C). Digital subtraction angiography (DSA) was performed and revealed bilateral CS DAVFs (Fig. 1D and E).
Fig. 1.
Diagnostic images of bilateral CS DAVFs. A: Enhanced MR images showing CS enhancement (frames in panels 1 and 2). B: MR angiography revealed abnormalities in the vessels (arrows). C: MR diffuse weighted images were normal. D: Anterior posterior and lateral DSA images of the left ECA (panels 1 and 2) and left ICA (panels 3 and 4) showing a left CS DAVF (arrows) supplied by the ECA and ICA and draining into the SPS and IPS. E: Anterior posterior and lateral DSA images of the right ECA (panels 1 and 2) and right ICA (panels 3 and 4) showing a right CS DAVF (arrows) supplied by the ECA and ICA and draining into the SPS and IPS. Abbreviations: CS: cavernous sinus; DSA: digital subtraction angiography; DAVF: dural arteriovenous fistula; ECA: external carotid artery; ICA: internal carotid artery; IPS: inferior petrous sinus; L: left; MR: magnetic resonance; R, right; SPS: superior petrous sinus.
Endovascular treatment (EVT) was performed under general anesthesia. Because DAVFs can be accessed via the pharyngeal trunk of the AphA (Fig. 2A), TAE via the pharyngeal trunk of the AphA was designed with the assistance of the “pressure cooker” technique. First, the pharyngeal trunk was catheterized with a Marathon microcatheter (Medtronic, Minneapolis, MI, USA). After the Marathon microcatheter closed the CS, the pharyngeal trunk of the AphA was coiled with two Prism coils sized 1.5–4 cm (Medtronic, Irvine, CA, USA) just below the Marathon microcatheter via an Echelon-10 microcatheter (Medtronic, Irvine, CA, USA), leading to the “pressure cooker” effect (Fig. 2B). Then, 6 ml of Onyx-18, a liquid embolic agent (Medtronic, Irvine, California, USA), was administered, obliterating most of the bilateral CS DAVFs (Fig. 2C–E). However, there was a small residual left DAVF (Fig. 2F). The residual DAVF was then obliterated by casting the Onyx via the left middle meningeal artery (MMA) and the accessory meningeal artery (AMA) (Fig. 2F–H).
Fig. 2.
EVT course of bilateral CS DAVFs. A: DSA images of the right ECA showing that the right PT (arrow) of the AphA was hyperplastic and could act via the transarterial route to embolize the DAVFs; the right NMT (arrow) origin was low. B: Left panel: X-ray image showing the coils in the PT; the arrow indicates the Marathon microcatheter tip. Right panel: Microcatheter angiography via the PT showing the fistula. C: X-ray images showing Onyx casting via the PT. D: DSA image of the right carotid artery showing satisfactory EVT. E: DSA image of the left carotid artery showing a residual fistula. F: DSA images of the left ECA showing microcatheter angiographies via the distal MMA and AMA (arrows). G: X-ray images (panels 1 and 2) and X-per computed tomography image (right panel) showing Onyx casting (arrows and frame) via the MMA and AMA. H: DSA image of the left carotid artery showing satisfactory EVT. Abbreviations: AMA: accessory meningeal artery; AphA: ascending pharyngeal artery; CS: cavernous sinus; DSA: digital subtraction angiography; DAVF: dural arteriovenous fistula; ECA: external carotid artery; EVT: endovascular treatment; L: left; MMA: middle meningeal artery; NMT: neuromeningeal trunk; PT: pharyngeal trunk; R, right.
The patient awoke postoperatively, had grade V muscle strength in the upper and lower limbs, and could walk normally. However, she suffered minor numbness and paralysis on the left side of her face. Postoperative 2nd-day computed tomography revealed that the Onyx agent had migrated into the intracranial vessels (Fig. 3A). Postoperative 3rd-day MR image revealed an acute infarction of the right head of the caudate nucleus (Fig. 3B). MR angiography and venography revealed no anomalies (Fig. 3C). One month later, the patient recovered well. Numbness and paralysis of the face disappeared.
Fig. 3.
Postoperative CT and MR examinations. A: Postoperative 2nd CT image showing Onyx migration into the intracranial vessels (arrows). B: Postoperative 3rd MR image revealing acute infarction of the right head of the caudate nucleus (arrows). C: MR venography (panels 1, 2 and 3) and angiography (panel 4) revealed no sign of a fistula. Abbreviations: CT: computed tomography; MR: magnetic resonance.
3. Discussion
Currently, for bilateral CS DAVFs, TVE is the first-line choice. In a 2013 meta-analysis, Perng et al. reported that, for the endovascular treatment (EVT) of bilateral CS DAVFs, the most common route for TVE was the trans-inferior petrous sinus (IPS), followed by the transfacial vein and OphV superior ophthalmic vein (OphV) (Fig. 4, Fig. 5) [5]. Because of the complex angioarchitectures of bilateral CS DAVFs, TVE is challenging. Fay et al. summarized the strategy of TVE for bilateral CS DAVFs. The general rule is to eliminate the symptomatic DAVF or the DAVF most likely to cause complications first. To manage low-flow bilateral CS DAVFs, a single session of TVE is often sufficient [3].
Fig. 4.
Trans-IPS EVT for bilateral CS DAVFs in a 56-year-old female. A: Panels 1 and 2: DSA images of the right ECA showing the right CS DAVF (arrow); venous drainage was extensive and involved the IPS, SPS and cortical veins (arrowheads). Panel 3: DSA of the right ICA showing that the DAVF drained via the SPS. B: Panels 1 and 2: DSA images of the left ECA showing the left CS DAVF (arrow); venous drainage involved the SPS and IPS; Panel 3: DSA of the left ICA showing that the DAVF drained via the cortical vein (arrowhead). C: Panel 1: Roadmap image showing that the right IPS (arrow) was catheterized; Panel 2: Microcatheter angiography showing that the microcatheter reached the left CS; Panel 3: X-ray image showing the Onyx casing in the entire CS. D-E: DSA images of carotid arteries showing that bilateral CS DAVFs had been obliterated. Abbreviations: CS: cavernous sinus; DAVF: dural arteriovenous fistula; DSA: digital subtraction angiography; ECA: external carotid artery; EVT: endovascular treatment; ICA: internal carotid artery; IPS: inferior petrous sinus; L: left; R, right; SPS: superior petrous sinus.
Fig. 5.
Trans-FV-OphV EVT for unilateral CS DAVFs. A: Hemorrhagic complication after EVT in a 58-year-old female: Panel 1: DSA images of the left ECA and ICA showing a CS DAVF (arrows) that drained via the OphV into the FV, and the cortical vein (arrowhead) was involved as the drainer; Panel 2: DSA image showing that the CS DAVF had been obliterated; Panel 3: Postoperative computed tomography showing subarachnoid hemorrhage, possibly due to venous drainage impairment. B: Successful EVT in a 57-year-old female: DSA images of the bilateral carotid arteries (panel 1) and left ECA (panel 2) showing a left CS DAVF (arrows) that drained via the left OphV into the FV; DSA images of the VA showing that the SCA supplied to the DAVF; Panel 4: Roadmap images showing that the FV and OphV were catheterized and that the microcatheter (arrow) reached the CS; Panel 5: X-ray image showing that the Onyx and coils packed the CS; Panel 6: DSA images of the bilateral carotid arteries and VA showing that the CS DAVF had been obliterated. After EVT, the patient experienced cranial nerve palsy, and after one year of follow-up, the cranial nerve palsy remained. Abbreviations: CS: cavernous sinus; DAVF: dural arteriovenous fistula; DSA: digital subtraction angiography; ECA: external carotid artery; EVT: endovascular treatment; FV: facial vein; ICA: internal carotid artery; L: left; OphV: ophthalmic vein; R, right; SCA: superior cerebellar artery; VA: vertebral artery.
Bilateral CS DAVFs may be isolated and have their own venous drainage. Bilateral CS DAVFs may also communicate via inter-CS channels, which flow into one side of the CS and its tributary [3]. Communicating bilateral CS DAVFs is common. For the communicating type of bilateral CS DAVFs, if only one transvenous approach can be used, the microcatheter should be navigated to the contralateral CS through the inter-CS channel; then, coiling can be performed from the contralateral CS to the ipsilateral CS. After coiling, if there is a residual fistula at high risk of venous drainage, Onyx can be administered in combination. Alternatively, spontaneous thrombosis of residual CS DAVFs can be anticipated.
Although TVE is the preferred approach for bilateral CS DAVFs, it can be difficult to coil all the shunt points, and the entire CS must be packed, increasing the risk of permanent cranial nerve palsy or the impairment of CS drainage. TAE is not the mainstream method for treating bilateral CS DAVFs and can only be attempted in select cases with hyperplastic feeding arteries. In our case, the shunt points of the bilateral CS DAVFs involved the entire CS, and routine transvenous routes were all occluded (Fig. 1D and E). However, there was a hyperplastic pharyngeal branch of the AphA as the feeding artery. Therefore, the pharyngeal branch may be chosen for embolization. Unlike the trans-MMA route, which involves dangerous anastomosis, the route via the pharyngeal branch of the AphA may be relatively safe because the pharyngeal branch is extracranial and does not supply cranial nerves.
TAE via the pharyngeal branch may be appropriate for treating CS DAVF or if there is an easily accessible AphA route [6,7]. For example, Fang et al. embolized 13 CS DAVFs via AphA, and 10 of the 13 fistulas were occluded successfully [8]. However, bilateral CS DAVFs treated by TAE via the pharyngeal trunk of the AphA have rarely been reported, highlighting the novelty of our approach. During TAE, the neuromeningeal trunk of AphA is not damaged by Onyx reflux. Therefore, the “pressure-cooker” technique with balloon assistance or coiling to occlude the feeding artery is useful and can limit Onyx reflux into the neuromeningeal trunk and increase the penetration of the Onyx.
TAE via AphA without the assistance of the “pressure-cooker” technique may be difficult for managing CS DAVFs due to the limited tolerable distance of Onyx reflux (Fig. 6) [9]. Because Scepter balloon catheter (Microvention, Tustin, California) was not available at our institute, we had to establish the “pressure cooker” effect by coiling the pharyngeal branch. After the pharyngeal branch was coiled to establish the “pressure-cooker” effect, most bilateral CS DAVFs were embolized (Fig. 2D and E).
Fig. 6.
Incomplete EVT for a CS DAVF via the AphA in a 60-year-old female. A: DSA images of the left carotid artery (panel 1), right ICA (panel 2) and right ECA (panel 3) showing a left CS DAVF (arrows) that drained into the cortical vein (arrowheads). B: Panel 1: DSA showing that AphA was hyperplastic; Panel 2: Microcatheter angiographies of the AphA showing the left CS DAVF (arrows); Panel 3: X-ray image showing Onyx casting in the CS and AphA. C: DSA images of the bilateral carotid arteries showing a small residual DAVF (arrows). D: DSA images of the bilateral carotid arteries at the six-month follow-up showing that the residual DAVF had disappeared. Abbreviations: AphA: ascending pharyngeal artery; CS: cavernous sinus; DAVF: dural arteriovenous fistula; DSA: digital subtraction angiography; ECA: external carotid artery; EVT: endovascular treatment; L: left; R: right.
During TAE via the AphA, to avoid complications associated with anastomoses of the AphA with external and internal carotid arteries and the vertebral artery, such as Onyx, which migrates into the intracranial vessels, the Onyx should be carefully injected via the AphA. These anastomoses are usually invisible on angiographies, but because of increased intra-arterial pressure during EVT procedures, they can open, resulting in stroke of the anterior and posterior circulations. In our case, during TAE, the Onyx migrated into the anterior and middle cerebral arteries through dangerous anastomosis (Fig. 3A). Fortunately, only asymptomatic acute brain infarction occurred (Fig. 3B). The route of Onyx migration was speculated from the superior pharyngeal branch of the AphA to the internal carotid artery (ICA) via the inferolateral trunk, the recurrent artery of the foramen lacerum or the clival branch of the meningohypophyseal trunk [10]. To prevent Onyx migration into the intracranial artery, a balloon in the cavernous ICA may be helpful for sealing the branches of the cavernous ICA.
After EVT, the patient experienced facial numbness and paralysis, both of which may have resulted from occlusion of the petrous branch and the artery of the trigeminal ganglion of the MMA to supply the facial nerve and trigeminal ganglion. The horizontal segment of the intra-temporal facial nerve is the most vulnerable because this segment occupies almost the entire intracanal space and can become ischemic once the intracanal pressure increases [11]. The blood flow of this portion is supplied by the petrosal branch of the MMA. Once the petrous branch is occluded, facial nerve injury can occur (Fig. 7). The recurrent branch of the AMA can supply the trigeminal ganglion, and its occlusion can result in trigeminal injury [12]. Based on the anatomy of the MMA and AMA, when casting Onyx via the MMA and AMA, the Onyx reflux should be controlled beyond the skull base level to avoid nerve injury.
Fig. 7.
Severe facial paralysis due to facial nerve injury after EVT for a CS DAVF via the MMA in a 68-year-old female. A: DSA images of the right internal carotid artery (left panel) and left external carotid artery (right panel) showing a CS DAVF (asterisks); the arrow indicates the petrous and cavernous branches of the MMA. B: Left panel: X-ray image showing the establishment of the “pressure cooker” effect by coiling the origin of the petrous and cavernous branches. The arrow indicates the Marathon microcatheter close to the DAVF. Right panel: Microcatheter angiography showing the fistula (asterisk). C: Left panel: After EVT, X-ray image showing the Onyx in the DAVF and petrous and cavernous branches of the MMA. Right panel: Reconstructive computed tomography image showing the Onyx (asterisk) in the posterior left CS. D: DSA images of the right carotid artery (left panel) and left carotid artery (right panel) showing that the CS was obliterated. Abbreviations: CS: cavernous sinus; DAVF: dural arteriovenous fistula; DSA: digital subtraction angiography; EVT: endovascular treatment; L: left; MMA: middle meningeal artery; R: right.
4. Conclusion
In select cases in which the hyperplastic pharyngeal branch of the AphA serves as the feeding artery, TAE can be performed to obliterate bilateral CS DAVFs with the assistance of the “pressure cooker” technique. However, possible stroke resulting from anastomoses of the AphA must be considered.
CRediT authorship contribution statement
Jinlu Yu contributed to data collection, writing-original draft, the manuscript review and revision.
Embolization of bilateral cavernous sinus dural arteriovenous fistulas via the ascending pharyngeal artery using the “pressure cooker” technique: A case report.
Consent for publication
Written informed consents were obtained from the patients for publication in this case report and any accompanying images. Written consents are available for review by the Editor-in-Chief of this journal.
Ethics approval and consent to participate
Ethics approval was not needed in our institution, as the manuscript was a case report.
Research registration number
Not applicable.
Funding
None.
Declaration of competing interest
The authors declare that they have no competing interests.
Acknowledgements
None.
Data availability
Data will be made available on request.
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Associated Data
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Data Availability Statement
Data will be made available on request.







