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|Year : 2014 | Volume
| Issue : 4 | Page : 393-399
Subarachnoid hemorrhage with blister aneurysms: Endovascular management
Swati Dayanand Chinchure1, Vipul Gupta1, Gaurav Goel1, Aditya Gupta2, Ajayanand Jha2
1 Department of Interventional Neuroradiology, Institute of Neurosciences, Medanta- the Medicity, Gurgaon, Haryana, India
2 Department of Neurosurgery, Institute of Neurosciences, Medanta- the Medicity, Gurgaon, Haryana, India
|Date of Submission||05-Mar-2014|
|Date of Decision||01-Jun-2014|
|Date of Acceptance||24-Aug-2014|
|Date of Web Publication||19-Sep-2014|
Department of Interventional Neuroradiology, Institute of Neurosciences, Medanta the Medicity, Gurgaon - 122 001, Haryana
Source of Support: None, Conflict of Interest: None
Blister aneurysms of are rare lesions representing a real challenge for diagnosis and management. They typically show small size, hemispherical shape, fragile wall, broad neck, and are arising from non-branching sites of intracranial arteries. Materials and Methods: We retrospectively reviewed all aneurysms treated at our institution. Seventeen patients (6 male, 11 female) with 17 blister aneurysms were identified (mean age 53.3, range 41-63 years). Clinical, procedural, angiographic data as well as follow up data were evaluated. Results: All patients presented with aneurysmal subarachnoid hemorrhage. Majority of the blister aneurysms were located in ICA while 1 was located at posterior cerebral artery, 1 at vertebral and 1 involving basilar artery. All patients were treated using single or overlapping stents and if possible additional coiling. There was no intra-operative rupture. Good outcome (mrs 0-2) was seen in 14 patients. Poor clinical outcome (mrs 3-5) was seen in 2 patients due to vasospasm induced ischemic deficits at discharge, both of them improved on follow up (mrs 1 on follow up). There were 3 mortalities, One patient died of rebleeding while other 2 died due to SAH induced complications. Follow-up angiography was available in 16 patients (one patient died before follow up angiogram) and revealed complete or near complete aneurysm occlusion in 11, incomplete obliteration in 1 and no change in 2 cases. Two cases showed post-treatment angiographic aneurysm recurrence. Both cases were managed with repeat coiling and overlapping stent placement. Conclusion: Endovascular management using single/overlapping stent and if possible coil placement is technically safe and feasible in blister aneurysms. Overlapping stents lead to better aneurysm occlusion than a single stent. Blister aneurysm in dorso-medial ICA showed higher tendency of continued growth/recurrence, higher incidence of clinical vasospasm and in these cases early angiographic follow-up is advisable. Repeat treatment should be considered promptly if necessary.
Keywords: Blood-blister like aneurysms, stent reconstruction, subarachnoid hemorrhage
|How to cite this article:|
Chinchure SD, Gupta V, Goel G, Gupta A, Jha A. Subarachnoid hemorrhage with blister aneurysms: Endovascular management. Neurol India 2014;62:393-9
| » Introduction|| |
Blister aneurysms (BA) are typically described as hemispheric aneurismal bulge protruding from non-branching sites from the intracranial arteries. , The term "blister" was introduced in 1988 by Takashi.  The most common site is dorsomedial wall of the supraclinoid segment of the internal carotid artery (ICA); uncommonly they are seen in anterior communicating artery and basilar artery.  Because of their small size, fragile-thin wall and a broad-based neck, these aneurysms are a real challenge to manage. Patients typically present with acute subarachnoid hemorrhage (SAH). The angiographic diagnosis of BA may be difficult because of their small size and irregular morphology.  Repeat angiography often documents luminal changes or growth of these lesions.  Their natural history remains unpredictable with the potential to increase in size, change configuration, and rebleed rate higher than saccular aneurysms. 
The walls of BA are extremely fragile and easily lacerated during surgery resulting in high rate of intraoperative rupture with need of parent artery occlusion in some cases during surgical repair. , Endovascular treatment almost always necessitates single/overlapping stent placement with need for anti-platelet therapy and its associated issues in patient with SAH. Irrespective of the modality of BA treatment (endovascular/surgical), there is risk of aneurysm regrowth and recurrent bleeding, and repeat treatment might be required. A recently conducted systematic literature review  of all reports of BA till 2013 showed that independent of the treatment method used, mortality of BA is high. The surgical morbidity for BA is estimated to be 21%, with a mortality of 17%. For endovascular outcomes, the systematic review found a morbidity of 3.4% and a mortality of 11.5% for BA.  There is lack of follow-up data in the literature with paucity of large case series due to rarity of this vascular lesion. There are few reports of endovascular management in the literature with very few series of more than 10 cases report on endovascular management of these aneurysms. ,, We report our experience in endovascular management along with clinical and technical outcomes of 17 consecutive cases of BA treated at our center.
| » Materials and Methods|| |
We performed retrospective review of the records of 548 consecutive patients treated at our center. Out of these, 17 BA in 17 patients (6 males and 11 females, mean age 53.3, range 41-63 year) were included. The inclusion criteria include (1) BA located at non-branching sites and (2) SAH corresponding to the aneurysm. Clinical characteristics, procedural and angiographic data as well as follow-up data were retrospectively evaluated.
All patients were admitted to the neurosurgery critical care unit. After complete clinical evaluation, patients underwent cerebral angiogram in biplane digital subtraction angiography (Axiom Artis Zee; Siemens, Erlangen, Germany). Three-dimensional rotational angiography was done in all cases. After the angiogram, the case was discussed between endovascular and neurosurgery teams. The patient and family were then counseled regarding the treatment options. If endovascular management was planned, loading dose of double anti-platelets (300 mg Ecosprin (Aspirin) +300 mg Plavix (Clopidogrel)/50 mg Prasugrel) was given in 2-4 hours before the procedure. If external ventricular drainage (EVD) was required, it was done before giving these medications and a post-procedure computed tomography (CT) scan was done to document appropriate position of drainage catheter and for any possible bleed. All procedures were performed under general anesthesia. A 6-F guiding catheter was placed into the corresponding distal cervical ICA. Under road map guidance, the stent delivery microcatheter (Prowler select plus, Covidien, Irvine, USA) was placed in the parent vessel distal to the aneurysm. Thereafter, if possible another microcatheter (Echelon 10, Covidien, Irvine, USA/Excelsior SL 10, Boston Scientific, Ireland) was placed at the base of the aneurysm and the stent (Enterprise, Codman, Raynham, MA, USA) was deployed to trap the aneurysm microcatheter. To avoid the risk of rupture caused by shifted catheter tip, few loops of coil were deployed before stent placement. Then coiling was performed with bare coils. In BA as it was too small and too shallow to allow for coils to be introduced, we planned single/overlapping stent placement. Proper heparinization was maintained during the procedure (ACT > 300). Intra-arterial Abxicimab (maximum up to 10 mg) was given if clots were seen on the stent struts on angiographies (done on 5- to 10-minute intervals for at least 30 minutes). Post-procedure Angio CT (Dyna CT, Siemens) was done in all cases to exclude bleeding during procedure.
We performed early check angiogram in all the cases of BA before discharge. After the procedure, double anti-platelets were continued for at least 6 months followed by 150 mg aspirin alone, which is continued indefinitely. In patients with symptomatic vasospasm, intra-arterial (IA) vasodilatation was done using Nimodipine and Milrinone.
| » Results|| |
[Table 1] illustrates the patient demographics and [Table 2] illustrates endovascular treatment with clinical and angiographic follow-up data. Seven patients were treated in acute (0-3 days after ictus), 8 in subacute period (3-13 days) and 2 in chronic period (14 days after ictus). Four patients presented with poor clinical grade (Hunt and Hess grade III-V); Fisher grade III SAH was seen in 11 patients. Majority of the aneurysms were located in ICA (dorsomedial wall of supraclinoidal ICA in 8 patients and in paraclinoidal in 6) while 1 was seen at posterior cerebral artery (PCA), 1 at vertebral artery and 1 involving basilar artery. Overlapping stents were placed in 5 patients while single stent was placed in 12 patients. Additional coils were placed in 6 Blister aneurysms; all of them were located at the dorsomedial wall of supraclinoid ICA.
One patient (Case 9) had cervical ICA dissection during 3D rotational angiography using pressure injector leading to complete ICA occlusion. Immediate placement of 2 overlapping carotid stents resulted in complete recanalization with normal antegrade flow. Overlapping intracranial stents were placed in supraclinoid ICA BA in the same sitting. Patient achieved complete clinical recovery. Another patient (case 12) developed small non-occlusive dissection in petrous ICA adjoining guiding catheter tip. Stent was placed across the dissection and patient did not have any complications because of the vessel injury. One patient (case 15) had dissection of left vertebral artery leading to complete occlusion and small posterior circulation infarction during previously done angiography at other hospital. In 2 patients thrombus formation was seen soon after stent placement, which resolved completely after intra-arterial abciximab.
Seven patients needed IA vasodilatation; six of them had BA located at dorsomedial wall of supraclinoid ICA. EVD was placed in 7 patients as per the protocol described above. During EVD removal, platelet transfusion was given. One patient (Case 5) developed patchy hemorrhages along EVD track, multiple episodes of nasal and gastrointestinal bleeding secondary to previous esophageal varices, and vision loss in one eye secondary to vitreous hemorrhage. One of the anti-platelet doses was stopped and he made complete recovery on follow-up. One patient developed EVD-induced meningitis and expired (Case 12).
The degree of aneurysm occlusion after coiling was classified into 3 categories: complete occlusion, near complete aneurysm occlusion (minimal neck remnant), and incomplete aneurysm occlusion (residual filling within the aneurysm perimeter/coil interstices) [Table 2]. Complete aneurysm obliteration after initial procedure was seen in 5 patients, out of which sustained aneurysm occlusion on follow-up was seen in 3 and aneurysm regrowth was seen in 2 patients (Cases 12 and 17). Both patients with aneurysm regrowth had complete aneurysm occlusion after initial procedure and underwent repeat treatment with additional stent and coil placement [Figure 1].
|Figure 1: Case 17: A 45-year-old female patient with acute SAH. Cerebral angiogram showed wide neck supraclinoid ICA blister (a). Stent-assisted coiling was done achieving near total aneurysm occlusion (b and c). Follow-up angiogram after 10 days showed signifi cant regrowth of the aneurysm (d). Another stent was placed across the neck of the aneurysm|
and aneurysm sac was embolized with coils (e) achieving complete aneurysm occlusion (f)
Click here to view
After the initial procedure incomplete aneurysm occlusion was seen in 5 patients, all of them on follow-up angiogram showed complete/near complete aneurysm occlusion [Figure 2]. Unchanged morphology of the BA after initial procedure was seen in 7 patients, out of which 5 patients showed progressive aneurysm occlusion [Figure 3] and 2 of them showed similar unchanged morphology at 4-month and 1-year angiographic follow-ups. Both these aneurysms with unchanged morphology were located in ICA in which single stent without coils had been placed. Both patients refused further treatment. One patient died without check angiogram (case 14).
|Figure 2: Case 13: A 62-year-old female with acute Fisher grade III SAH (a). 3D rotation cerebral angiogram and corresponding DSA showed wide neck supraclinoid ICA blister (b and c). Stent-assisted coiling was done achieving near complete aneurysm obliteration. Arrows are pointing to stent markers (d). Follow-up angiogram after 10 days (e) showing|
complete aneurysm occlusion
Click here to view
|Figure 3: Case 4: A 41-year-old man with acute SAH. Cerebral angiogram showed wide neck blister aneurysm arising from dorsomedial wall of supraclinoid ICA on right side (a). Stent-assisted coiling was attempted but coil could not be placed. Note the tortuous anatomy of cervical ICA (b). Double overlapping stent was placed across the neck of the aneurysm. Post-procedure angiogram showed no change in the morphology of the aneurysm (c). Follow up angiogram after 1 year|
showed complete aneurysm occlusion (d)
Click here to view
At initial presentation of 13 patients showed good clinical grade (Hunt and Hess 0-II) while 4 patients were in poor clinical grade (Hunt and Hess III-V). In the group of 13 patients with good clinical grade at admission, at discharge good clinical outcome (modified Rankin score 0-2) was seen in 12 patients; one patient died of rebleeding after complete aneurysm occlusion (Case 17). Among the 4 patients with poor clinical grade at admission, 2 patients showed good clinical outcome on follow up (modified Rankin score 0-2) while the remaining 2 expired, one with EVD-induced meningitis (case 12) and another because of cardiopulmonary arrest during chest procedure (case 14).
| » Discussion|| |
BA is different from saccular aneurysm. Saccular aneurysms are true aneurysms, with the walls formed by thickened intima and/or adventitia, which is composed of collagenous tissue. In BA, there is focal gap in the internal elastic lamina and media, which is covered with thin lacerated- fragmented adventitia and clot suggesting pseudoaneurysm. , Associated focal sub-adventitial dissection at rupture site has been reported in 40% to 89% of the cases. , Angiographic differentiation between BBA and saccular aneurysms is difficult. Hemispherical shape and location at non-branching site may help in differentiating it from true saccular aneurysms. However, few reports showed that with continued growth and with absorption of clot at rupture point, the BA may take a saccular shape. ,
For pseudoaneurysm and hemorrhagic dissection, parent artery occlusion either by surgical and by endovascular trapping may be the most definite means to prevent rebleeding provided there is sufficient collateral circulation.  Balloon test occlusion in the setting of acute SAH is usually not possible, and even in those tolerant of Balloon test occlusion, delayed ipsilateral cerebral ischemia after ICA trapping was reported. , This might be because of the subsequent development of severe vasospasm compromising collateral circulation. Parent artery occlusion may also interfere with endovascular access for subsequent vasospasm treatment.
Different surgical options have been proposed to address BA, including direct clipping, clipping plus wrapping, wrapping alone, clipping with Sundt encircling graft clips, encircling silicone clip application, primary suturing of ICA, vascular staple clip closure of ICA, and trapping with or without extracranial-intracranial bypass. ,,, High intra-and postoperative bleeding have been reported during surgical clipping indicating difficulties arising in operative management. ,, Recently published surgical and long-term follow-up data in BA by Kalani and group  showed intraoperative rupture in 4 out of 17 cases during attempted clipping, 2 cases (out of 17) had postoperative bleeding and 1 had angiographic recurrence after clipping. The rebleeding risk for BA is higher than for saccular aneurysms, and peri-operative hemorrhage can be life-threatening and significant. ,
Endovascular management is also technically challenging because of the weak nature of BA and small size of the aneurysms with a broad neck. Different endovascular options described in the literature are single/overlapping stenting/stent assisted coiling and flow diverter placement. A stent placement is essential in most cases so as to retain the coils within the aneurysm sac. Stent not only acts as new flow conduit within that vessel altering flow dynamics within the aneurysm but also provides matrix for intimal growth reducing the likelihood of recurrence.  Theoretically, the overlapping stents at the aneurysm neck would create better flow diversion than a single stent and decreases hemodynamic stress effectively.  Stent placement necessitates anti-platelet therapy, which can be a management issue in patients with recent SAH. Moreover, these patients may require ventricular drainage secondary to hydrocephalus and can develop hemorrhage along catheter track. Stent-assisted coil embolization has been applied in treating BA in several centers. ,,,,, Attempt to pack the aneurysm completely with coils may result in intraoperative rupture , and loose packing is likely to result in continued growth of aneurysm. ,, Placement of another overlapping stent in subsequent sittings after initial treatment by stent assisted coiling is reported to prevent regrowth in BA.  In our observation, endovascular treatment is quite safe in these difficult aneurysms with low risk of complications after use of double anti-platelets. We did not have any bleeding during the procedure. By carefully planning the strategy for ventricular drainage as mentioned, majority of the cases were managed without significant complications related to anti-platelets. We had only one case of bleeding along the drainage track (Case 5) who ultimately made good clinical recovery.
We observed 3 cases of spontaneous dissections during angiography (Cases 9, 12, 15), 2 dissections happened during diagnostic angiography and one during intervention. All the angiographies were performed by formally trained neurointerventionists. All of these patients had BA located at the dorsomedial wall of supraclinoid ICA, probably suggesting that some of these patients may have propensity to dissect and any possibility of underlying connective tissue disorder should be considered. Extreme care is recommended during angiography of these cases. Two of our cases with paraclinoidal BA treated with single stent (Cases 1, 6) showed no changes in morphology on follow-up. In our experience, double overlapping stents lead to better aneurysm occlusion rates than single stents on follow-up. Second, we observed higher rate of aneurysm growth as well as repeat bleeding in BA located at the dorsomedial wall of supraclinoid ICA. Therefore, it may be a better strategy to plan for overlapping stents in these locations as well as in coil placement whenever possible. Recent availability of modern ultrasoft and smaller coils has led to better and probably safer aneurysm occlusion. However, early angiographic follow-up is mandatory. On follow-up, Parent artery occlusion can be considered if aneurysm shows angiographic regrowth provided the patient has good collaterals.
Flow-diverter stents may offer a new option for the treatment of difficult small BA. By redirecting blood flow along the normal course of the parent artery, flow-diversion devices alter dynamic fluid exchange across the aneurysm/parent vessel interface, creating an intra-aneurysmal environment that is conducive to thrombosis. The construct then becomes progressively incorporated into the parent artery through a process of neo-intimal overgrowth. , Available data of limited cases ,, show promising short-term results, although there are issues of the post-procedural intracranial hemorrhage, side branch/perforator occlusion and stent thrombosis. Flow diverters are highly thrombogenic and when placed in supraclinoid ICA, it can overlap anterior cerebral artery origin, which can create further clinical issues during subsequent vasospasm. Questions remain regarding long-term safety, treatment results and long-term clinical outcomes. Better understanding of the devices and continued research is needed. Cost remains a major issue, especially in Indian subcontinent.
To the best of our knowledge, till date this is probably the largest series of BA treated by endovascular route. A limitation of this report is that it is a single-center experience with small study size, lack of a comparative control group and retrospective evaluation. Although our case series suggests a beneficial treatment outcome from the treatment algorithm studied (single stent/overlapping double stent/stent-assisted coiling), further prospective evaluation with larger patient populations is warranted.
In conclusion, BA is difficult to treat. Endovascular management using single/overlapping stent, and if possible coil placement, is technically safe and feasible. Overlapping stents lead to better aneurysm occlusion than a single stent placement. Issues related to anti-platelet usage can be handled in most cases with carefully planned protocols. BA in dorsomedial wall of ICA showed higher tendency of continued growth/recurrence and higher incidence of clinical vasospasm; in these cases, early angiographic follow-up is advisable. Repeat treatment should be considered promptly if necessary. In future, larger studies are needed to compare these results with flow diverters.
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[Figure 1], [Figure 2], [Figure 3]
[Table 1], [Table 2]
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