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Show Pipeline Embolization Device: A New Source for Embolic Retinal Vascular Occlusion Adam B. Sise, MD, James M. Osher, MD, Martin P. Kolsky, MD, Andrew Stemer, MD, William O. Bank, MD, Richard A. Garfinkel, MD Abstract: A 57-year-old woman underwent treatment of a left internal carotid artery aneurysm with a Pipeline embolization device. She subsequently experienced multiple branch retinal artery occlusions in her left eye. Although rare, ophthalmic complications may follow this new technique in the treatment of intracranial aneurysms. Journal of Neuro-Ophthalmology 2013;33:373-376 doi: 10.1097/WNO.0b013e3182a304d5 © 2013 by North American Neuro-Ophthalmology Society The Pipeline embolization device (PED; ev3/Covidien, Irvine, CA) has emerged as a novel endovascular treat-ment for intracranial aneurysms. It is a cylindrical braided wire mesh implant that creates a pathway to bypass aneurys-mal cavities. By diverting blood flow through the implant, blood in the aneurysmal cavity is left to stagnate leading to thrombosis, thus walling it off from the parent vessel. Approved by the Food and Drug Administration in 2011, it has shown success in treating intracranial aneurysms, espe-cially those with challenging anatomic subtypes (1-4). Although there have been rare reports of vision loss related to the device, we describe a patient who presented with multiple branch retinal artery occlusions (BRAOs) following PED placement for treatment of an intracranial aneurysm of the internal carotid artery. CASE REPORT A 57-year-old woman was evaluated for left-sided headache, neck pain, and excess lacrimation from the left eye. Her medical history was significant for hypertension, and she had a brother who died 4 years previously from complica-tions related to a subarachnoid hemorrhage. Magnetic resonance imaging (MRI), magnetic resonance angiography (MRA), and computed tomography angiography revealed a 6.8 · 6.4 · 6.8-mm3 aneurysm at the anterior genu of the left internal carotid artery, opposite to the origin of the left FIG. 1. Three-dimensional reconstructed computed tomo-graphic angiogram shows an aneurysm of the left internal carotid artery (arrow). The origin of the ophthalmic artery is not visible in this projection. Department of Ophthalmology (ABS), Georgetown University Hospi-tal, Washington, District of Columbia; Medstar Washington Hospital Center (ABS, JMO, MPK), Washington, District of Columbia; The Retina Group of Washington (JMO, RAG), Chevy Chase, Maryland; Cincinnati Eye Institute (JMO), Cincinnati, Ohio; and Neurointerventional Service (AS, WOB), Medstar Washington Hospital Center, Washington, District of Columbia. The authors report no conflicts of interest. Informed consent was sought and granted for publication of this report. Address correspondence to James M. Osher, MD, Washington National Eye Center, 110 Irving Street NW, Suite 1A-1, Washington, DC 20010; E-mail: osher.james@gmail.com Sise et al: J Neuro-Ophthalmol 2013; 33: 373-376 373 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. ophthalmic artery (Fig. 1). The patient was treated with 2 Pipeline devices telescoped from the origin of the left posterior communicating artery to the vertical segment of the petrous portion of the left internal carotid artery cover-ing the entire length of the aneurysm (Fig. 2). There were no reported intraoperative or immediate postoperative com-plications, and postprocedure angiography of the parent and branching vessels appeared normal (Fig. 3A). The patient was discharged on clopidogrel and aspirin. Three weeks after the procedure, she presented to the emergency department with sudden, painless vision loss in her left eye. Emergent computed tomography, MRI, and MRA of the head and neck revealed no acute abnormal-ities, expected artifact from the Pipeline device, and no evidence of aneurysmal filling (Fig. 4). The patient refused cerebral angiography. Neuro-ophthalmic examination dis-closed visual acuity of 20/25, right eye, and 20/40, left eye. Color vision was impaired in the left eye, and there was a left relative afferent pupillary defect. Eyelids, slit-lamp examination, and extraocular movements were normal. The right fundus was unremarkable while the left showed area of retinal whitening along the superotemporal and inferotem-poral arcades, which contained plaque material. Cotton wool spots were seen temporal and superonasal to the optic disc (Fig. 5). Fluorescein angiography revealed delayed filling of the superotemporal arteriole with eventual retrograde perfu-sion (Fig. 6). Platelet inhibition at the time of the patient's vision loss was measured using the VerifyNow point-of-care system (Accumetrics, San Diego, CA). P2Y12 and aspirin reaction units were within normal limits. The patient was diagnosed with multiple BRAOs and instructed to continue her aspirin and clopidogrel. Cerebral angiography performed during routine 6-month follow-up showed patent flow through the Pipeline devices and complete obliteration of the aneurysm, with some narrowing of the proximal Pipeline device at its proximal and distal ends (Fig. 3B). The left ophthalmic artery filled through the walls of the Pipeline devices, and there was an intact choroidal blush of the left eye. The patient's visual acuity stabilized at 20/20 bilaterally, with a dense visual field defect in the right eye. DISCUSSION The PED represents a significant improvement in the treatment of intracranial aneurysms compared with conven-tional microsurgical techniques in both aneurysmal occlusion rates and patient outcomes (4,5). The PED is placed within the parent artery and, because of this, carries a risk of throm-boembolic complications until endothelialization is complete. Most Pipeline procedures are performed with double anti-platelet therapy to decrease these risks because aneurysmal occlusion occurs over many months (5,6). The Pipeline for Uncoilable or Failed Aneurysms (PUFS) trial was the major clinical study that led to Food and Drug Administration approval and included safety and efficacy data FIG. 2. Lateral skull radiograph shows the Pipeline devices (arrows) within the left internal carotid artery immediately after placement. FIG. 3. Lateral view of subtracted cerebral angiogram. A. Immediately after Pipeline device placement, there is faint contrast blush (arrow) within the aneurysm. The ophthalmic artery (arrowhead) is patent. B. Six months later, there is no filling of the aneurysm, and the ophthalmic artery remains patent. Arrows denote the proximal and distal ends of the second Pipeline device where there is mild narrowing. 374 Sise et al: J Neuro-Ophthalmol 2013; 33: 373-376 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. on 108 patients (1). Although our patient's aneurysm was smaller than the criteria used in the PUFS study, a PED was used in our patient because of the wide neck of the aneurysm. The PUFS report included 5 cases of amaurosis fugax, none of which occurred in direct relationship to the procedure. The other 4 occurred after Day 90 and there was no evidence of retinal arteriole occlusion. There was 1 case of cilioretinal artery occlusion that occurred on the day of the procedure (1). Several subsequent large studies failed to report perma-nent visual loss due to embolic arteriole occlusion (4-11). In our patient, it is presumed that the narrowing of the second Pipeline device may have caused a disruption of flow into the ophthalmic artery, or thrombus, that embolized distally into the retinal arterioles. The cause of the narrowing is unclear, and although the use of angioplasty to widen the areas of stenosis was considered, ultimately this was dismissed because of the patient's stable clinical course. Long-term data are still needed to fully evaluate the risk of delayed events related to Pipeline device placement, and patients should be appropriately counseled regarding the risk of vision loss both during and in the months after the procedure. REFERENCES 1. U.S. Food and Drug Administration. Pipeline embolization device-P100018. Summary of safety and Effectiveness data (SSED). Available at: http://www.accessdata.fda.gov/cdrh_ docs/pdf10/P100018b.pdf. Accessed April 6, 2011. 2. Nelson PK, Lylyk P, Szikora I, Wetzel SG, Wanke I, Fiorella D. The pipeline embolization device for the intracranial treatment of aneurysms trial. AJNR Am J Neuroradiol. 2011;32:34-40. 3. Lylyk P, Miranda C, Ceratto R, Ferrario A, Scrivano E, Luna HR, Berez AL, Tran Q, Nelson PK, Fiorella D. Curative endovascular reconstruction of cerebral aneurysms with the Pipeline embolization device: the Buenos Aires experience. Neurosurgery. 2009;64:632-642. 4. Sughrue ME, Saloner D, Rayz VL, Lawton MT. Giant intracranial aneurysms: evolution of management in a contemporary surgical series. Neurosurgery. 2011;69:1261-1271. 5. Yu SC, Kwok CK, Cheng PW, Chan KY, Lau SS, Lui WM, Leung KM, Lee R, Cheng HK, Cheung YL, Chan CM, Wong GK, Hui JW, Wong YC, Tan CB, Poon WL, Pang KY, Wong AK, Fung KH. Intracranial aneurysms: midterm outcome of Pipeline embolization device-a prospective study in 143 patients with 178 aneurysms. Radiology. 2012;265:893-901. 6. Kan P, Siddiqui AH, Veznedaroglu E, Liebman KM, Binning MJ, Dumont TM, Ogilvy CS, Gaughen JR Jr, Mocco J, Velat GJ, Ringer AJ, Welch BG, Horowitz MB, Snyder KV, Hopkins LN, Levy EI. Early postmarket results after treatment of intracranial aneurysms with the Pipeline embolization device: a US multicenter experience. Neurosurgery. 2012;71:1080-1088. 7. Leung GK, Tsang AC, Lui WM. Pipeline embolization device for intracranial aneurysm: a systematic review. Clin Neuroradiol. 2012;22:295-303. FIG. 4. Reconstructed magnetic resonance angiography. Susceptibility artifact is seen along the Pipeline device within the left internal carotid artery, with flow-related signal along its length. The supraclinoid internal carotid artery is patent with normal caliber. Flow-related signal is present within the left ophthalmic artery (arrow). FIG. 5. The left fundus shows areas of retinal whitening with plaques within the superotemporal and inferotemporal arterioles (arrows). Cotton wool spots (arrowheads) also are present. FIG. 6. Fluorescein angiogram of the left eye during laminar phase. Arrows denote areas of visible plaque. There is marked filling delay of the superotemporal vessels but the inferior vessels fill normally. Sise et al: J Neuro-Ophthalmol 2013; 33: 373-376 375 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. 8. Fargen KM, Velat GJ, Lawson MF, Mocco J, Hoh BL. Review of reported complications associated with the Pipeline embolization device. World Neurosurg. 2012;77:403-404. 9. Chitale R, Gonzalez LF, Randazzo C, Dumont AS, Tjoumakaris S, Rosenwasser R, Chalouhi N, Gordon D, Jabbour P. Single center experience with pipeline stent: feasibility, technique, and complications. Neurosurgery. 2012;71:679-691. 10. Szikora I, Berentei Z, Kulcsar Z, Marosfoi M, Vajda ZS, Lee W, Berez A, Nelson PK. Treatment of intracranial aneurysms by functional reconstruction of the parent artery: the Budapest experience with the pipeline embolization device. AJNR Am J Neuroradiol. 2010;31:1139-1147. 11. McAuliffe W, Wycoco V, Rice H, Phatouros C, Singh TJ, Wenderoth J. Immediate and midterm results following treatment of unruptured intracranial aneurysms with the pipeline embolization device. AJNR Am J Neuroradiol. 2012;33:164-170. 376 Sise et al: J Neuro-Ophthalmol 2013; 33: 373-376 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. |