Giant Basilar Artery Aneurysm in the Adolescent Patient: Case Report and Literature Review

Issabayev D.O., Surdin D.A., Makhambetov Y.T., Olenbay G.Y

Pediatric Neurosurgery , Pediatric endovascular surgery, Kazakhastan 

*Corresponding author

Issabayev D.O, Pediatric Neurosurgery , Pediatric endovascular surgery, Kazakhastan.

Introduction

Giant basilar artery (BA) aneurysms are uncommon. Frequently, aneurysms with a diameter of at least 25 mm have atheromatous plaques and are partially thrombosed. There are some problems in the treatment of giant aneurysms, especially when the aneurysm is localized in the posterior circulation, because ischemia of affected brain area might be caused by clipping or coil embolization. In addition, there are no guidelines for antiplatelet therapy in case of flow divert stenting within child and adolescents’ population.

A high predisposition for rupture is a characteristic feature of posterior circulation aneurysms. The cumulative 5-year risk of rupture is 2.5% for aneurysms smaller than 7 mm and 15% for aneurysms equal to or larger than 7 mm. [2] In cases of rupture, posterior circulation aneurysms have a particularly inauspicious prognosis, with a survival rate of only 32% at 48 hours. [3]

Giant aneurysms are some of the rarest and the most formidable lesions that neurosurgeons encounter, these lesions, which account for 2-3% of all aneurysms, are defined by the "Cooperative Study of Intracranial Aneurysms and Subarachnoid Hemorrhage" as aneurysms with a diameter of at least 2.5 cm. [3] The prognosis for patients with untreated giant aneurysms is might be unfavorable [4,5], thereby the safe and total obliteration of the aneurysm should be the aim of treatment. Historically, giant aneurysms have been treated by open surgery. These days, endovascular techniques have enabled clinicians to make great strides and improvements in the treatment of these difficult cases, but the long-term quality of these techniques remains a concern.

Children are found to have giant aneurysms (25 mm) far more frequently than adults. However, there is a large range in the numbers, from 0% to 68%. The numbers, however, vary widely from an incidence of zero to 68% [6, 7, 8, 9, 10, 11, 12, 13, 14, 15]. Krishna et al. [16] reported the incidence of giant aneurysms in children to be 13,6%, whereas the corresponding number in adults was 6,5%. Meanwhile, Proust et al. [9] found a frequency of giant aneurysms around 14% in both pediatric and adult patients. In infants <1 year, the mean size of aneurysms was 18mm, with 30/131 being giant [17]. Pediatric giant aneurysms seem to favor the vertebrobasilar arteries with reported incidences in that specific location ranging from 8-100% [18, 19, 20, 21, 22, 23, 24].

Nowadays, the treatment of giant aneurysms had become safer for patients, as minimally invasive technologies have progressed significantly in the practice of endovascular surgery, allowing for new treatment strategies , such as flow-divert stenting or endovascular coilng or balloon-assisted coiling is performed. When endovascular coiling fails, surgical treatment with bypass surgery and aneurysm trapping is a possible treatment modality for BA cases, but it requires surgical experience and has a high rate of complication. Here we present a rare case of a patient with a symptomatic giant aneurysm of the middle third of the BA, who treated successfully by coil trapping method.

Case Report

This 14-year-old male presented with chronic headache and left convergent strabismus. MRI/MRA showed the aneurysm in the middle part of the basilar artery (BA) with a brainstem mass effect (Figs. 1).

Figure 1: MRI shows evidence giant aneurysm of the basilar artery with mass effect.
A - frontal view.
B - axial view.
C - sagittal view.

Figure 2: Left vertebral artery (VA) diagnostic cerebral angiography shows a wide-neck giant saccular aneurysm (25.9 x 12.7 x 18 mm, with a neck of 6 mm) in the middle part of the BA aneurysm.
A: Frontal view.
B: Sagittal view.

Figure 3: A: View from the left side. White arrows show the stent placement in the middle third of the basilar artery.
B: Frontal view. In the arterial phase, there are signs of contrast agent stagnation in the aneurysm cavity. O’Kelly–Monrotta scale A.

Figure 4: Representation of blood flow changes due to stent migration.
A: Red arrows represent the flow of blood through the stent. Green color shown migration of proximal part of the stent.
B: Green arrows represent the flow of blood through the stent.

Figure 5: Tried to insert microcatheter into the first stent via PcomA was failed. Both PcomA are functioning.
A: frontal view. B: left-side view.

Figure 6: A - Disconnecting the aneurysm from the blood flow was supported by using a stent migration. B - Coil trapping at the level of the middle third of the basilar artery

Figure 7: Successful coil trapping of the middle third of the basilar.
A: Frontal view. B: Left-side view. C: From the left ICA fills the upper third of BA, and the aneurysm is not contrasted.

Figure 8: Upper left - MRI before operation. Down left - MRI TOF after operation. Upper right - Follow up in 1 year after operation (TOF). Down right - MRI TOF before operation.

Diagnostic angiography of the left vertebral artery (VA) was notable for a prominent right and left posterior communicating artery(PcomA) coupled with an atretic right A1 segment of the right anterior cerebral artery(ACA), and arterial-phase images of the left VA were notable for a giant saccular aneurysm of middle 1/3 of basilar artery (BA) with size of 25,9 x 12,7 x 18 mm, with a wide neck of 6 mm.(Fig.2)

In this case, due to the involvement of the middle part of the BA and potential mass effect, microsurgical treatment or endovascular coiling alone was not indicated. Consequently, we made the decision to deploy a flow diverter stent in front of the aneurysm neck in the BA.

The patient received dual antiplatelet therapy (ticagrelor 180 mg/d and aspirin 100 mg/d) 2 days before the surgery.

Surgery day

A flow-diverting stent Silk Vista Baby 3,0mm*20mm was deployed in the middle third of the basilar artery in front of aneurysm neck. (Fig 3)

Follow-Up in six months

Two months after flow diversion stent deployment, the strabismus and headache subsided. At 6-month follow-up, diagnostic cerebral angiography showed no aneurysmal shrinkage. Migration of the proximal part of the stent toward the aneurysm neck and changes in bloodstream between the stent body and the wall of the basilar artery are shown (Fig. 4).

Due to stent migration, we decided to place a second stent telescopically into the first stent. (Fig 5).

Using contralateral angiographic study of the ICA by filling the posterior circulation via the PCommA, the adequacy of collateral circulation was assessed by compression of the carotid artery in the neck with the fingers (Matas test). Consequently, it has been proven that cerebral blood flow in the posterior circulation is completely compensated by the PCommA.

Considering favorable anatomic variant of the both posterior communicating arteries and the stent migration into the aneurysm that had created a stable frame for optimized packing of coils, we had decided to coil embolization aneurysm and trapping the middle third of the basilar artery (Fig. 6).

Control cerebral angiograms from the VA showed filling of the lower third of the basilar artery; from the left ICA showed filling of the cerebral blood flow of the right and left posterior cerebral arteries and left and right superior cerebellar arteries(SCA) through the PCommA without signs of slowing down the passage of the contrast agent to the capillary phase.(Fig.7)

There were no complications in the early and late postoperative period. The patient was discharged for ambulatory observation without neurological deficit.

Follow-Up in 1-year after surgery

Compared with preoperative MRI, MRA one year after surgery showed no filling of the aneurysm sac due to thrombosis and no mass effect from the aneurysm (Fig 8).

Discussion

Common risk factors for the development and rupture concerning all intracranial aneurysms, counting basilar artery aneurysms,2 include female sex, family history, connective tissue disease, past SAH and hypertension. Vertebrobasilar aneurysms account for 10% to 18% of all intracranial aneurysms, and 50% to 65% of these lesions occur in the basilar bifurcation. Although not as common as aneurysms of the anterior circulation, lesions of the posterior circulation seem to be associated with higher risks and worse prognosis. Another large meta-analysis examining both patient and aneurysm characteristics as risks for rupture found an increased relative risk of rupture 2.5 for aneurysm’s in the posterior circulation, one of the valuable predictors for rupture class with increasing size and symptomatic lesions [25, 26].

This has been borne out for both unruptured and ruptured aneurysms in several studies. Regarding the former, the International Study of Unruptured Intracranial Aneurysms (ISUIA) found that posterior circulation aneurysms of all sizes had a higher 5-year rate of rupture compared with similar size of cavernous and anterior circulation aneurysms. A Japanese single institution series also found a hazard ratio of 2.9 for unruptured aneurysms of the posterior circulation, a similar trend demonstrating the increased risk of rupture for this location [27].

The use of flow-diverting stents is likely to further improve the outcome of patients with giant aneurysms, but data on long-term outcomes for these devices are still pending, and their use may not be the optimal treatment modality in all cases. [28, 29] Despite significant advancements in the neurovascular field, the substantial information and reported outcomes for the safe and total obliteration of giant aneurysms remain from surgical cases.

Nevertheless, it has been proposed that the outcomes of cerebral flow diversion may not be applicable to children due to basic differences between intracranial aneurysms in adults and children [30]. Although endovascular treatment with flow diverters has shown good angiographic results, high complete occlusion rates [31, 32] and acceptable overall procedure-related morbidity in adults, there is a higher risk of unfavorable outcomes in posterior circulation aneurysms and giant aneurysms, which are more frequently encountered in the pediatric population [33].

Based on our results, we believe that flow diverters can significantly enhance the endovascular treatment of cerebrovascular disorders. However, in present case, the discrepancy in stent size—since there was no equivalent stent-size available at the time of the operation—caused the proximal part of the stent to migrate into the aneurysm cavity. So, we do not have in order sufficient length of the stent to be hooked into the current vessel.

Therefore, several issues still require further clarification.

The first issue is children’s growth. The ongoing growth of cerebral vessels in children has been identified as a potential pitfall by some authors [34, 35] or even as a contraindication for cerebral stent placement or flow diversion in this age group [36]. According to Waitzman et al, cranial growth is rapid during the first year of life, slows markedly in later years, and is almost complete by age 6 [37]. Arat et al 28 showed that by 48 months of life, 81%–99% of adult diameters were attained in intracranial arteries; however, only 59% of adult diameters were attained for the common iliac artery [38]. Later, similar findings regarding children were also reported by He et al. From a morphometric standpoint, these studies suggest the following: The size range of current intracranial stents or flow diverters is sufficient to cover the pediatric population, and intracranial arterial diameters in children do not undergo striking growth, especially after early childhood. Thus, the implication that stent placement could cause in intracranial stenosis or similar complications later in life is not supported by the current literature [39].

The second issue that concerns us the most is the pre-/post-procedural antiplatelet medication protocols as there is no standard antiplatelet/anticoagulant therapy for children undergoing intracranial placement of vascular scaffolds (ie, stents, stent grafts, or flow diverters). Reviewing 35 case reports on the placement of cerebral vascular scaffolds in children, we discovered that the use of antiplatelet agents for endovascular treatment was extremely variable [35]. There are neither guidelines published on antiplatelet therapy (APT) in children with cerebrovascular diseases 25 nor conclusive trials on pediatric APT regimens [40, 41].

In this case, due to the constitutional characteristics of the patient weighing >60 kg and the child being >14 years of age, we decided to use 180 mg Ticagrelor per day prior to surgery as with the adult dosage.

Endovascular coiling has been deemed the most effective treatment for aneurysms of this location due to its better patient outcomes than surgical treatment as had shown in randomized clinical trials [41]. However, endovascular coiling alone is not always feasible for complex and/or fusiform aneurysms, even if stent-assisted coiling or balloon-assisted coiling is performed [42]. If endovascular coiling fails, surgical treatment with bypass surgery and aneurysm trapping is a possible treatment modality for these complex BA cases, but it requires surgical experience and has a high rate of complications [43].

But in this case, localization in the posterior circulation presented a great difficulty in the form of a high risk of possible complications. Our clinic uses a trapping method for giant aneurysm of ICA’s. [44] Due to the presence of both posterior communicating arteries, we decided to direct trapping of the middle third of the basilar artery.

Drake et all. performed trapping of the involved segment of the basilar artery in two of 13 patients with aneurysms of the giant basilar trunk [46]. 7 cases in his series were treated with Gunther ligation of the basilar artery due to basilar artery aneurysms with or without rupture, and 5 cases experienced 50% success in aneurysm thrombosis without a significant neurological deficit.

Conclusion

If the anatomical variant is favorable, especially the presence of two posterior communicating arteries, coil trapping of the basilar artery can be performed for giant aneurysms. In our case, coil trap at the level of the middle third of the basilar artery and disconnecting the aneurysm from the blood flow was supported by using a stent migration.

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