AUTHOR(S): Guglielmi, Guido, M.D.; Viñuela, Fernando, M.D.; Briganti, Francesco, M.D.; Duckwiler, Gary, M.D. Department of Neurological Sciences, Service of Therapeutic Neuroangiography, School of Medicine, University of Rome (GG), Rome, Italy, and Department of Radiological Sciences, Endovascular Therapy, School of Medicine, University of California (FV, FB, GD), Los Angeles, California Neurosurgery 31; 591-596, 1992 ABSTRACT: A CASE OF carotid-cavernous fistula caused by a ruptured intracavernous aneurysm is reported. The fistula was treated with electrothrombosis by the detachment of two platinum coils into the aneurysm via an endovascular transvenous approach; the fistula was closed, and the patient has recovered completely. The advantages of using electrodetachable platinum coils include thrombogenicity, controllable deposit, radiopacity, and biocompatibility. KEY WORDS: Carotid-cavernous fistula; Detachable coils; Electrothrombosis; Interventional neuroangiography; Intracranial aneurysm Carotid-cavernous (c-c) fistulas of the direct type caused by the rupture of an intracavernous aneurysm are uncommon (1,9). At present, endovascular treatment by the detachment of a balloon into the fistula is the recommended treatment for direct-type cc fistulas (1-3,8,13,14). In case of failure of the detachable balloon embolization procedure, the use of platinum coils to be enlarged into the fistula via a transvascular route has been suggested (6-8,15). This report refers to a direct-type c-c fistula caused by a ruptured intracavernous aneurysm treated by transvascular electrothrombosis with detachable platinum coils; the features and the advantages of this technique are discussed. CASE REPORT A 28-year-old woman sought treatment for the acute onset of severe headache, left eye proptosis, and orbital pain. Computed tomographic scans and magnetic resonance imaging revealed enlargement of the left cavernous sinus. A left carotid angiogram depicted a left c-c fistula, with a major venous drainage through the ipsilateral inferior petrosal sinus (Fig. 1). A vertebral angiogram obtained during left carotid compression allowed visualization of a left intracavernous carotid aneurysm that had spontaneously ruptured, thus producing the c-c fistula

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(Fig. 2a). To treat the fistula, an endovascular treatment with detachable balloons was planned. Via a transfemoral approach, under systemic heparinization, a transfemoral temporary balloon occlusion of the left internal carotid artery (ICA) was performed by the inflation of a balloon proximally to the fistula. During a 30-minute occlusion, the electroencephalographic recording and the clinical examination showed no modifications. In spite of several attempts, it was not possible to introduce a detachable balloon into the fistula through a transarterial approach because of the small size of the fistulous communication. Alternatively, through a transfemoral venous approach, an 8 French catheter was positioned into the left internal jugular vein; through it, a Tracker-18 microcatheter (Target Therapeutics, San Jose, CA) was introduced into the cavernous sinus and then into the ruptured aneurysmal sac (Fig. 2b). A 20-cm-long platinum detachable coil (Guglielmi Detachable Coil; Target Therapeutics) was then introduced through the microcatheter into the aneurysm. The detachable coil consists of a soft platinum coil soldered to a stainless steel delivery wire; the entire stainless steel wire is covered with an insulating material, with the exception of a short distal portion. Electrothrombosis was then elicited by the application of a 0.5 mA/2 V positive direct current to the proximal end of the stainless steel delivery wire; the negatively charged red and white blood cells, platelets, and fibrinogen were therefore attracted toward the positively charged intraaneurysmal platinum coil. By the time a thrombus formed around the platinum coil, the uninsulated stainless steel portion detached immediately inside the aneurysm. The same procedure was then repeated to detach another 15-cm-long platinum coil into the aneurysm (Fig. 3). During the delivery of both coils, to prevent their migration toward the arterial stream, a nondetachable balloon, positioned at the level of the aneurysmal neck, was temporarily inflated into the left ICA; the occlusive balloon was removed from the left ICA after the delivery of the second coil. An immediate postembolization angio-gram demonstrated a slightly decreased flow through the cc fistula (Fig. 4). Heparinization was reversed by the administration of protamine sulfate to the patient. A 3-day follow-up angiogram indicated that thrombosis of the arteriovenous communication had occurred. A 3-month control angiogram did not depict any further change. Contrast opacification of the portion of the aneurysm that had not been filled with coils was present (Fig. 5). Proptosis and ocular pain progressively regressed and eventually disappeared 1 week after the embolization; the subsequent clinical course was uneventful. DISCUSSION The rupture of an aneurysm of the intracavernous portion of the ICA generally leads to the sudden onset of a high-flow c-c fistula (1) with an evident clinical symptomatology, mainly represented by

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Neurosurgery 1992-98 September 1992, Volume 31, Number 3 591 Carotid-Cavernous Fistula Caused by a Ruptured Intracavernous Aneurysm: Endovascular Treatment by Electrothrombosis with Detachable Coils Technical Note

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The recent development (4) of soft platinum coils attached to a stainless steel carrying wire, electrolytically detached distally by the application of a direct electrical current at the proximal end of the guidewire, can overcome some of the abovementioned problems. Moreover, the electrical current in the wire creates a positive charging of the distal platinum coil, thus inducing electrothrombosis by attracting the negatively charged red blood cells, white blood cells, platelets, and fibrinogen (electrothrombosis) (4). Electrothrombosis by means of these detachable soft platinum coils via the endovascular approach has been recently performed in selected cases of intracranial aneurysms (5). The experimental and clinically applied techniques have been extensively described (4,5). The transvenous approach was chosen in this case because the small fistulous communication probably would have prevented the successful detaching of coils by the transarterial route, as it did for the detachable balloon procedure. It was also possible to temporarily inflate a nondetachable balloon in the ICA at the level of the fistula to prevent inadequate migration of transvenous coils into the arterial stream. The detachable and retrievable coils used in this case could be easily manipulated into a proper position, giving them a satisfactory shape before the release. Had migration or unsatisfactory position been observed, the coil could have been completely withdrawn and repositioned into the aneurysm. The coils are soft enough to adapt to the shape of the aneurysmal sac without significantly increasing the intra-aneurysmal pressure. When each platinum coil was thought to be in a suitable position, electrothrombosis was elicited by the application of a low-voltage, direct positive current to the proximal end of the delivery guidewire; therefore, the positively charged platinum coil would attract the negatively charged blood components, thus inducing thrombus formation. Then, by electrolysis, in a few minutes, the current dissolved the uninsulated stainless steel junction zone between the guidewire and the coil, so that each coil was detached into the aneurysm. An immediate thrombosis was prevented probably by the systemic heparinization during the procedure. Subsequently, intraaneurysmal and intracavernous thrombosis progressed with time, so that in the 3-day control angiogram the fistula was occluded. Although residual opacification of the aneurysm occurred because the coils did not entirely fill the sac, the embolization with detachable platinum coils might be repeated, if complete filling of the aneurysm is mandatory. In this case, the procedure was not repeated, because complete closure of the fistula occurred. The advantages of the electrodetachable platinum coils are related to their biocompatibility, radiopacity, and thrombogenicity; they may be controlled by retrieval and repositioning. Their malleability and the possibility of detachment with no need for pulling help prevent mechanical trauma to the surrounding

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proptosis, ocular pain, congestion of ocular bulb vessels ("red eye"), impairment of vision, subjective and objective retrobulbar bruit (generally, direct-type c-c fistulas result from closed head trauma, with/without basal skull fractures, although they also occur without apparent precipitating factors in collagen deficiency diseases, in vessel dissections, from iatrogenic injuries, and after the rupture of intracavernous carotid aneurysms [7]). In recent years, intravascular embolization with detachable balloons has emerged as the treatment of choice for direct-type c-c fistulas (1-3,8,13,14). In 1988, Halbach et al. (6) reported the results of 165 direct-type c-c fistulas treated by means of endovascular therapy: in 14 patients (8.5%), the embolization was performed through a transvenous approach because of the failure of transarterial attempts, previous carotid artery occlusion or trapping, or overwhelming risks related to collagen deficiency disease. Also, the transarterial approach may not be successful if the volume of the venous compartment is too small to allow balloon inflation or if the arteriovenous fistulous communication is too small to permit entry of the balloon (6). Through the transarterial approach, the placement of standard, nonattached platinum coils into the cavernous sinus is an alternative treatment when balloon embolization fails (7,15). A number of problems related to the use of this technique are reported: the coil(s) alone can be inadequate to produce a thorough occlusion because of low thrombogenicity and the impossibility of obtaining a suitable shaping into the fistula. However, soft coils made of platinum mixed with dacron fibers seem to carry an increased thrombogenicity. Coils may also be impacted in the microcatheter while being pushed into position through the catheter. Another major disadvantage of the treatment of the direct-type c-c fistulas with platinum coils is represented by the fact that loss of the coil during its deposition is possible, in that an unexpected distal migration of the coil immediately after the delivery may take place, with untoward embolization in the distal vascular tree or in the draining vein(s), respectively (7). Furthermore, once a coil is placed into the delivery catheter, it cannot be retrieved. Intraoperative electrothrombosis of c-c fistulas and aneurysms induced by platinum or copper needle electrodes has been described (10-12); in these, the thrombogenicity of the wire was enhanced by the application of direct electrical current. Mullan (11) reported six cases of c-c fistulas in which thrombosis was obtained by means of platinum or copper electrodes inserted with either stereotactic technique or direct surgical exposure; in all cases, total occlusion of the fistula with preservation of the parent vessel was achieved. Hosobuchi (10) reported the treatment of four cases of direct-type c-c fistulas by means of intra-operative electrothrombosis with copper wires and needles; the wires and needles were inserted into the draining sinus through cannulation of the superior ophthalmic vein or by craniotomy. In all cases, he obtained closure of the fistula, with preservation of the internal carotid circulation.

12.

ACKNOWLEDGMENT This work was supported by grant NEUA 1 RO1 HL46286-01 from the National Institutes of Health.

13.

Received, November 18, 1991. Accepted, April 3, 1992. Reprint requests: Guido Guglielmi, M.D., UCLA Medical Center, 10833 Le Conte Avenue, B2-188, CHS 172115, Los Angeles, CA 90024.

14. 15.

REFERENCES: (1-15) 1.

2.

3.

4.

5.

6.

7.

8.

9.

10. 11.

Barrow DL, Spector RH, Braun IF, Landman JA, Tindall SC, Tindall GT: Classification and treatment of spontaneous carotid-cavernous fistulas. J Neurosurg 62:248-256, 1985. Debrun DB, Lacour P, Vinuela F, Fox A, Drake CG, Caron JP: Treatment of 54 traumatic carotid- cavernous fistulas. J Neurosurg 55:678-692, 1981. Debrun DM, Vinuela F, Fox AJ, Daves KR, Ahn KS: Indication for treatment of 132 carotid-cavernous fistulas. Neurosurgery 22:285-289, 1988. Guglielmi G, Vinuela F, Sepetka I, Macellari V: Electrothrombosis of saccular aneurysms via endovascular approach. Part 1: Electrochemical basis, technique and experimental results. J Neurosurg 75:1-7, 1991. Guglielmi G, Vinuela F, Dion J, Duckwiler G: Electrothrombosis of saccular aneurysms via endovascular approach. Part 2: Preliminary clinical experience. J Neurosurg 75:8-14, 1991. Halbach VV, Higashida RT, Hieshima GB, Hardin CW, Yang PJ: Transvenous embolization of direct carotid-cavernous fistulas. AJNR 9:741- 747, 1988. Halbach VV, Higashida RT, Barnwell SL, Dowd CF, Hieshima GB: Transarterial platinum coil embolization of carotidcavernous fistulas. AJNR 12:429-433, 1991. Higashida RT, Halbach VV, Tsai FY, Norman D, Pribram HF, Mehringer CM, Hieshima GB: Interventional neurovascular treatment of traumatic carotid and vertebral artery lesions: Results in 234 cases. AJR 143:577-582, 1989. Higashida RT, Halbach VV, Barnwell SL, Dowd C, Dormandy B, Bell J, Hieshima GB: Treatment of intracranial aneurysm with preservation of the parent vessel: Results of percutaneous balloon embolization in 84 patients. AJNR 11:633-640, 1991. Hosobuchi Y: Electrothrombosis of carotidcavernous fistula. J Neurosurg 42:76-85, 1975. Mullan S: Experiences with surgical

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thrombosis of intracranial berry aneurysms and carotic cavernous fistulas. J Neurosurg 41:657-670, 1974. Nishijima M, Kamiyama K, Oka N, Endo S, Takaku A: Electrothrombosis of spontaneous carotid-cavernous fistulas by copper needle insertion. Neurosurgery 14:400-405, 1984. Serbinenko FA: Balloon catheterization and occlusion of major vessels. J Neurosurg 41:151- 154, 1974. Vinuela F, Dion J, Lylyk P, Duckwiler G: Update on interventional neuroradiology. AJR 153:23- 33, 1989. Yang PG, Halbach VV, Higashida RT, Hieshima GB: Platinum wire: A new transvascular embolic agent. AJNR 9:447450, 1989.

COMMENTS Hosobuchi and Mullan (1,2), as slightly older neurosurgeons will remember, successfully occluded carotid cavernous fistulas with electrotherapy. Because this was not always effective and because it usually required surgery, other avenues were explored. Balloons became the device of choice for curing direct carotid cavernous fistulas (indirect carotid cavernous fistulas are those associated with "dural arteriovenous malformations"). They did not always work, but improved navigational abilities came with newer catheters and guidewires to which the balloons could be directly attached, allowing precise control and placement. Still, there are some failures. Microcoils became useful, especially those with dacron "feathers," now used to block the venous side of "unballoonable" fistulas. Guglielmi and his UCLA colleagues add another indication for the use of his Guglielmi Detachable Coil, which is an ingenious coil placed through a microcatheter and used to electrically thrombose the fistula (or aneurysm), and which itself is then detached electrolytically. These coils are more malleable than microcoils, are controllable until detachment, and will be the treatment of choice for many fistulas and intracranial aneurysms. Although all of the "bugs" are not out, work continues at a rapid pace at the centers using these devices. Mullan and Hosobuchi were right, and for the right reasons, but were ahead of their technology. David O. Davis Washington, District of Columbia REFERENCES: (1,2) 1. 2.

Hosobuchi Y: Electrothrombosis of carotidcavernous fistula. J Neurosurg 42:76-85, 1975. Mullan S: Experiences with surgical thrombosis of intracranial berry aneurysms and carotid cavernous fistulas. J Neurosurg 41:657-670, 1974.

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structures. These features allowed endovascular treatment of a direct-type c-c fistula, in which the embolization with a detachable balloon was not possible.

Alex Berenstein New York, New York

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Dr. Guglielmi and coauthors have introduced a detachable electrocoil that represents a most significant advancement in endovascular therapy. The ability to introduce an endovascular device into the vascular system, verify its position, and if its not optimal, retrieve it, reposition it, or replace it for a different size--and be able to detach these platinum coils at will in a controlled and reproducible manner-is analogous to the placement of a vascular clip in a control fashion. The use of electrothrombosis to enhance occlusion and electrolysis to detach the device has been used with very encouraging results in the treatment of large, difficult aneurysms, by the authors, and 10 additional groups. In this report, the authors are describing the use of the same device in the management of a carotid-cavernous fistula secondary to a spontaneous rupture of an internal carotid artery aneurysm and have been able to treat both lesions. The flexibility of the system is further demonstrated by its ability to navigate from the venous system to the arterial site and occlude not only the fistula but also the aneurysm that caused it. We have confirmed the reliability of Guglielmi Detachable Coil devices on a vertebrovertebral fistula, where balloons were unable to enter the fistulous tract, and therefore, I would further venture to say that these devices may replace detachable balloons in the management of a variety of cerebral vascular lesions by the endovascular route when the parent vessel is to be preserved. The only point of caution would be the effect of the electrothrombosis on the cranial nerves in the region of the cavernous sinus; however, the ability to detach the coil at will, even after it has been extruded in the vascular system, makes this technique a reliable and reproducible one.

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Figure 1. Left carotid angiogram (lateral projection) showing a left c-c fistula mainly draining through the inferior petrosal sinus (arrow).

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Figure 2. A, Vertebral angiogram obtained during left carotid compression (lateral projection); the left supraclinoid (arrows) and cavernous portions of the ICA fill in a retrograde fashion via the posterior communicating artery (arrowhead), thus showing the fistula and the intracavernous aneurysm (curved arrows) (see Text). (B) Intra-aneurysmal (arrows) contrast injection (lateral projection) through a microcatheter introduced into the aneurysmal sac via a transvenous approach; the inferior petrosal sinus is also visible (double arrows).

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Figure 3. Plain x-ray film (lateral projection) showing two platinum coils (total length 35 cm) detached inside the aneurysm (arrows).

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Figure 4. Immediate postembolization left internal carotid angiogram (lateral projection) showing the coils inside the aneurysmal sac (arrows); the flow through the fistula is only slightly diminished.

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Figure 5. Three-month follow-up left internal carotid angiogram (lateral projection). The fistula is completely occluded, whereas the ICA is patent. The portion of the aneurysm not filled with coils is still visible (arrow). A 3-day follow-up angiogram (not shown) provided the same findings.

Carotid-cavernous fistula caused by a ruptured intracavernous aneurysm: endovascular treatment by electrothrombosis with detachable coils.

A case of carotid-cavernous fistula caused by a ruptured intracavernous aneurysm is reported. The fistula was treated with electrothrombosis by the de...
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