Abstract BACKGROUND Intracavernous aneurysms constitute up to 9% of all intracranial aneurysms and 6% are infectious (IIA). First line therapy is a protracted antibiotic course, yet with failure, surgery and endovascular parent vessel sacrifice have been utilized. Reconstructive endovascular therapies have emerged for aneurysm control and may demonstrate a safer therapeutic alternative. OBJECTIVE To present an IIA treated with a flow-diverting Pipeline stent (ev3 Neurovascular, Irvine, California). METHODS A 41-yr-old female presented with visual loss, ophthalmoplegia, and cavernous sinus thrombosis with an associated phlegmon. Transsphenoidal evacuation was performed without complication or bleeding and she continued on medical therapy. Two weeks postoperatively, she developed a worsening right third cranial nerve palsy and MRA demonstrated a 1-cm right IIA, not evident on postoperative MRI. Three days of dual antiplatelet therapy preceded successful pipeline embolization. Angiography demonstrated aneurysm obliteration at 3 mo and her right ophthalmoplegia resolved. RESULTS A literature review identified 6 reported cases of IIAs treated with stent embolization. Only 1 documented a flow-diverting Silk stent used in a child. All lesions were obliterated at follow-up without neurological sequelae. No complication arose with implantation in the setting of infection, and as few as 3 d of dual antiplatelet therapy was sufficient for preprocedural prophylaxis, although in Vivo antiplatelet activity may be more significant. CONCLUSION We report the first case of an IIA treated with a flow-diverting pipeline stent. These devices preserve native vasculature and neurological function compared to surgical and endovascular vessel sacrifice strategies. They appear to be safe management options for the treatment of IIAs. Cavernous carotid, Flow-diversion, Infectious aneurysm, Mycotic aneurysm, Pipeline, Stent, Intracavernous carotid ABBREVIATIONS ABBREVIATIONS ACA anterior cerebral artery ICA internal carotid arteries LP lumbar puncture MRA magnetic resonance angiography MRV magnetic resonance venography PED Pipeline embolization device POD postoperative day Mycotic aneurysms constitute 2% to 9% of all intracranial aneurysms and usually occur at bifurcation points in the distal middle cerebral and anterior cerebral arteries.1 Intracavernous aneurysms constitute up to 9% of all intracranial aneurysms and of these, 2.5% to 6% are infectious.2-6 Untreated infectious aneurysms in this region can cause cranial neuropathies, cavernous carotid fistulas, and cavernous sinus thromboses.7–9 First line therapy of a long course of antibiotics may resolve lesions completely; however, refractory cases may require surgical or endovascular treatment.10 Flow-diverting stents are just starting to be reported as technically feasible treatment options that offer the advantage of both adjacent and parent vessel preservation with low associated morbidity.1 Only 1 previous case in a pediatric patient has been reported utilizing a flow diverting stent, the Silk device, for infectious aneurysms and our report is the first demonstrating the use of a Pipeline embolization device (PED; ev3 Neurovascular, Irvine, California) for the successful management of an infectious cavernous aneurysm in an adult patient.1 METHODS A 41-yr-old female with a past medical history of polycystic ovarian syndrome presented to the emergency department after waking up with complete loss of vision in the left eye, left-leg weakness, and progressive lethargy over the previous 2 wk. Upon admission to the emergency department, she was febrile to 102.3 and had leukocytosis to 18.9. Lumbar puncture (LP) showed significantly elevated leukocytes, glucose, and protein and negative cultures. On exam, she had bilateral proptosis with nonreactive pupils, complete bilateral third nerve palsy, and decreased left V1 and V2 sensation. MRI/magnetic resonance venography (MRV) demonstrated cavernous sinus and superior ophthalmic vein thrombosis with soft tissue filling of the sella and sphenoid sinuses (Figure 1A). Narrowing of both cavernous internal carotid arteries (ICAs) was noted, in addition to bilateral posterior cerebral venous infarctions. FIGURE 1. View largeDownload slide A, T1W with contrast and fat supression: Soft tissue within the right cavernous sinus (consistent with thrombosis) and with extension to the paranasal sinuses. In addition there is narrowing of the cavernous carotid arteries. B, CT obtained on postoperative day #1 demonstrating no hemorrhage or sign of vascular injury with a small amount of air in the postoperative cavity. FIGURE 1. View largeDownload slide A, T1W with contrast and fat supression: Soft tissue within the right cavernous sinus (consistent with thrombosis) and with extension to the paranasal sinuses. In addition there is narrowing of the cavernous carotid arteries. B, CT obtained on postoperative day #1 demonstrating no hemorrhage or sign of vascular injury with a small amount of air in the postoperative cavity. She was admitted to the intensive care unit and started on a broad spectrum antibiotics, amphotericin, a heparin drip, and high-dose dexamethasone. Repeat MRI 4 d later was concerning for a rapidly progressive infectious process involving the orbits and sella. Given the increasing sellar collection and inconclusive blood cultures, the patient was transferred to our institution for urgent surgical decompression. She underwent uncomplicated transsphenoidal resection of the suprasellar phlegmon, with no evidence of bleeding or arterial trauma. The carotid artery was not visualized and postoperative CT demonstrated no hemorrhage or mass in the sellar region and only a small amount of air adjacent to packing in the operative bed (Figure 1B). Her headaches improved but her left eye persisted without light perception and a lateral gaze palsy and her right eye exhibited complete third nerve palsy. On postoperative day (POD) 2, there was concern for continuing septic emboli given new bilateral cerebellar and anterior cerebral artery (ACA) territory infarcts on MRI. Cultures eventually grew gram-positive filamentous rods from the sellar lesion, actinomyces, and sinus cultures speciated aspergillus fumigatus. While she remained neurologically stable, follow-up MRI/magnetic resonance angiography (MRA) on POD 14 demonstrated increased cavernous sinus enhancement and ICA narrowing. A decision was made to undergo cerebral angiogram to further investigate the MRA findings, which demonstrated a new 13 × 12 × 5 mm right cavernous ICA aneurysm, with severe narrowing of cavernous and supraclinoid ICAs, along with bilateral A1-2 junction ACA narrowing, thought to be due to vasculitis. She was started on 325 mg of aspirin and 75 mg of Plavix (Bristol-Myers Squibb, New York, New York) daily and plan was made to undergo pipeline embolization of the infectious right cavernous aneurysm on POD 19. Endovascular Treatment Angiography demonstrated a slightly larger right cavernous ICA aneurysm (14.34 × 13.92 with a 5.3 mm neck) as well as slow filling within the aneurysm, suggesting progressive thrombosis (Figure 2). The venous phase demonstrated delayed venous outflow of the right superior ophthalmic vein consistent with persistent cavernous sinus thrombosis. Measurements of the ICA landing zones were obtained for proper sizing of the stent (Figure 2). The PED (ev3; 3 × 10 mm) was deployed across the neck of the aneurysm with control angiogram demonstrating slow and late filling of the aneurysm dome. A second PED (3.25 × 10 mm) was then deployed across the neck of the aneurysm. Proper stent placement was again confirmed with improving flow through the supraclinoid right ICA (Figure 3). The patient remained hemodynamically stable throughout the procedure and experienced no intraoperative complications. She was discharged 4 d later after medical and antibiotic optimization. Institutional Review Board approval was obtained and the patient consented for the presentation of her case upon follow-up care. FIGURE 2. View largeDownload slide A and B, Two-week postoperative lateral and frontal angiograms demonstrating new late-filling of the right cavernous carotid artery aneurysm and vasculitis of the bilateral anterior cerebral arteries. C, Three-dimensional aneurysm reconstruction demonstrating selected landing zones for the intended pipeline stent and vessel diameters at these points. FIGURE 2. View largeDownload slide A and B, Two-week postoperative lateral and frontal angiograms demonstrating new late-filling of the right cavernous carotid artery aneurysm and vasculitis of the bilateral anterior cerebral arteries. C, Three-dimensional aneurysm reconstruction demonstrating selected landing zones for the intended pipeline stent and vessel diameters at these points. FIGURE 3. View largeDownload slide A, Control angiogram demonstrating proper deployment of both pipeline stents across the aneurysm neck with slowed filling of the aneurysm. Note the stenosis of the ICA immediately distal to the stent. B, Unsubtracted lateral image demonstrating the location of the PEDs in relation to the adjacent subtracted images. C, Three-month follow-up angiogram demonstrating aneurysm obliteration and improved flow to the immediately distal carotid artery. FIGURE 3. View largeDownload slide A, Control angiogram demonstrating proper deployment of both pipeline stents across the aneurysm neck with slowed filling of the aneurysm. Note the stenosis of the ICA immediately distal to the stent. B, Unsubtracted lateral image demonstrating the location of the PEDs in relation to the adjacent subtracted images. C, Three-month follow-up angiogram demonstrating aneurysm obliteration and improved flow to the immediately distal carotid artery. RESULTS Three-month follow-up angiography showed complete obliteration of the right cavernous ICA aneurysm, with persistent narrowing of the right supraclinoid ICA with moderate improvement in vessel caliber and improved filling of right A1 segment from prior study (Figure 3). Mild narrowing consistent with intimal hyperplasia of the stent in the cavernous carotid was noted. Her vision remained poor in left eye, but her right ptosis and ophthalmoplegia improved. The paresis of her left leg resolved as well. DISCUSSION Osler first described a case of a mycotic aneurysm in 1885 by demonstrating an infectious pathological process in the walls of the vasculature of an autopsy specimen.11 By far, streptococcus and staphylococcus are the most common pathogens arising from bacterial endocarditis and meningitis, yet less than 50% of cases are culture positive.9,12 In our case, aspergillus was the pathogenic organism, reported previously in only a handful of cases.13 Males are twice as likely to developed mycotic aneurysms in this location and the mean age at presentation is 36-yr old, yet there is a wide range.10,13 The rate of aneurysm formation may vary from 24 h to 1 mo and given the propensity to either expand, resolve, or spread farther, serial surveillance has been advocated.14 Endovascular techniques of parent artery sacrifice with balloons, coiling, and liquid embolization have all been described with high degrees of exclusion of the aneurysms from circulation. More recently, the use of intracranial stents has become an emerging management strategy that allows for the preservation of the parent artery and the exclusion of the aneurysm from circulation.9 Micropore flow diverting stents are the newest additions to the armamentarium for the management of intracavernous stents and have been successful in maintaining parent vessel and adjacent vessel blood flow, while altering flow dynamics in the aneurysm and promoting neointimal growth over the surface of the stent itself to exclude the aneurysm. Both the Silk and Pipeline devices utilize this mechanism and are tailored specifically for intracranial use by allowing a high degree of flexibility to cover tortuous vasculature.15 To date, 6 reports to our knowledge in the English literature have documented endovascular stent placement for the treatment of intracavernous mycotic aneurysms, with only 1 documenting the use of a flow-diverting stent, the Silk device.1,11,13,14,16,17 Neuroform stents have been used in 3 patients who have initially failed medical management, with complete obliteration of all aneurysms at follow-up. The cardiac stent, the Helistent (Hexacath, Rueil-Malmaison, France), has also been used by 1 group who felt that the opening force of this cardiac stent was sufficient for a patient in whom concurrent stenosis adjacent to an intracavernous aneurysm was apparent. The Aneugraft PCS (Amnis Life, Or Akiva, Israel), another cardiac stent has also been used successfully to resolve a mycotic aneurysm in a child after 4 d of antiplatelet therapy. The final report described the successful use of a Silk flow diverter in a 10-yr old who failed medical management and balloon test occlusion, with no untoward events and obliteration of the aneurysm at 3 mo. The PED has also been used to treat pseudoaneurysms in the ICA with complete resolution of all aneurysms reported at follow-up.18,19 The reported cases all documented trauma or operative manipulation of the vasculature. While our team discussed the possibility that our case represented a pseudoaneurysm secondary to surgery in the sellar region, it is certain that there was not any injury to the carotid artery during transsphenoidal washout of the phlegmon as there was no evidence of substantial bleeding, or exposure that could make the carotid artery susceptible to injury. Additionally, the surrounding cavernous thrombosis was highly suggestive of infectious spread, and no hemorrhage was noted on postoperative CT. Several considerations arise when managing infectious aneurysms endovascularly, including the use and duration of dual antiplatelet therapy, the implantation of a device in the setting of infection, the delay to surgical management to trial a sufficient course of antibiotic therapy, and the choice of optimal management strategy. In regard to antiplatelet therapy, the risks of hemorrhage or further hemorrhage in ruptured cases must be carefully weighed against the need for protecting the stent and parent vessel from thrombosis. In some patients, this risk is unavoidable, and must be weighed against the risk of stroke in parent artery sacrifice techniques. In unruptured cases, 3 to 4 d may be sufficient for preprocedural antiplatelet therapy for stent placement as ours and other reports have demonstrated good initial results. Evaluation of antiplatelet activity with quantitative assays such as thromboelastography allows for the direct measurement of antiplatelet efficacy and may be more important in determining the safety of stent use in the acute setting. These studies have become part of the routine care of all patients undergoing stent embolization at our institution. Regarding the use of an implant in an infectious case, there have been no reports of graft infection in the literature. It remains unclear if infection may spread to the graft if antibiotics are not trialed first; however, it would contradict the standard of care to not trial antibiotics immediately upon diagnosis. In regard to the spontaneous resolution of some lesions and the persistence of others with antibiotic therapy, to date no associated factors have been identified to determine which aneurysms resolve with medical therapy. Antibiotic trials have been advocated in the literature unless new symptoms or evolving lesions become apparent, and these trials appear safe in stable patients as a first line approach. Our report, among others, demonstrates that stent placement is sufficient to obliterate aneurysms from circulation in this region, does not involve direct manipulation of friable aneurysm walls, and minimizes the risk of hypoperfusion of distal tissue, although this risk is not absent. While still off-labeled use, flow-diverting stents appear safe in this region and are advantageous over covered stents in their hypothetical maintenance of flow to small side branches, despite a lag in aneurysm obliteration compared to covered stents, which has not been shown to be clinically significant. Based on the 2 cases of flow-diverting stents used to treat mycotic aneurysms (this case included), these devices appear to offer safe, viable options for the management of mycotic cavernous aneurysms, which should be investigated further in the future. CONCLUSION We report the first case of a mycotic cavernous aneurysm treated with a PED (ev3), a newer flow-diverting stent used to protect flow to adjacent side branches off the parent artery, exclude flow to the aneurysm by changing flow dynamics within it, and maintain flow through the parent artery. This case adds to our understanding of safety and utility of these devices, and demonstrates treatment options that preserve as much of the native circulation as possible, compared to older strategies. Disclosure The authors have no personal, financial, or institutional interest in any of the drugs, materials, or devices described in this article. Notes An abstract of the current manuscript was presented as a poster at the 2017 American Association of Neurological Surgeons annual meeting in Los Angeles, California, April 22 to 26, 2017. REFERENCES 1. Appelboom G, Kadri K, Hassan F, Leclerc X. Infectious aneurysm of the cavernous carotid artery in a child treated with a new-generation of flow-diverting stent graft: case report. Neurosurgery . 2010; 66( 3): E623- E624. Google Scholar CrossRef Search ADS PubMed 2. Stiebel-Kalish H, Kalish Y, Bar-On RH et al. Presentation, natural history, and management of carotid cavernous aneurysms. Neurosurgery . 2005; 57( 5): 850- 857. Google Scholar CrossRef Search ADS PubMed 3. Eguchi T, Nakagomi T, Teraoka A. Treatment of bilateral mycotic intracavernous carotid aneurysms: case report. J Neurosurg . 1982; 56( 3): 443- 447. Google Scholar CrossRef Search ADS PubMed 4. Todo T, Inoya H. 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Google Scholar CrossRef Search ADS PubMed Copyright © 2017 by the Congress of Neurological Surgeons This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/about_us/legal/notices)
Operative Neurosurgery – Oxford University Press
Published: Aug 5, 2017
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