| Distal Lenticulostriate Artery Aneurysm Presenting With Spontaneous Intracerebral and Intraventricular Hemorrhage: A Case Report and a Review of the Literature. |
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Sung, Jae Kyung , Koh, Hyeon Song , Kang, Chang Woo , Kwon, Hyon Jo , Youm, Jin Young , Kim, Seon Hwan |
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Department of Neurosurgery, Chungnam National University School of Medicine, Daejeon, Korea. kohhs@cnu.ac.kr |
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| Abstract |
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The authors report here on a rare case of aneurysm involving the distal lenticulostriate artery (LSA) in a 66-year-old man who presented with intracerebral hemorrhage (ICH) in the right basal ganglia and also intraventricular hemorrhage (IVH). Three-dimensional computed tomography angiography (3D-CTA) and conventional cerebral angiography showed a 4 mm, round-shaped aneurysm in the right distal LSA and this was combined with moyamoya-like disease. We performed proximal clipping of the aneurysm using a microsurgical technique and we evacuated the hematoma. After the operation, there was recurrent bleeding around the operation site and hydrocephalus gradually developed, and we implanted a ventriculo-peritoneal (V-P) shunt. The patient did well after the final shunt surgery and rehabilitation. Presently, he has no motor weakness or significant neurologic deficit, but mild cognitive dysfunction remains. When spontaneous ICH occurs in an unusual site, a thorough investigation is important to rule out a structural vascular abnormality. |
| Key Words:
Aneurysm, Distal lenticulostriate artery, Hemorrhage |
Introduction
Spontaneous intracerebral hemorrhage (ICH) is reported in about 10~20% of all strokes,4)7) and it frequently causes significant neurologic deficits. Many of these hemorrhages are related to microaneurysms that form in patients with hypertension and they are rarely discovered by cerebral angiography. Aneurysms arising from the branches of the distal lenticulostriate artery (LSA) are a rare cause of spontaneous ICH; there are about 25 case reports of distal LSA aneurysms in the medical literature. Due to its rarity, we report here a case of a ruptured distal LSA aneurysm that presented as spontaneous ICH and intraventricular hemorrhage (IVH) in a healthy male patient, and we review the relevant literature.
Description of the case
A 66-year-old man was transferred from a local clinic with a continuous intractable headache that had started three days previously. He had no history of hypertension, diabetes or taking any specific medication and he had no definite neurologic deficit. A brain computed tomography (CT) scan showed a right basal ganglia ICH and IVH (Fig. 1-A, 1-B). Three-dimensional CT angiography (3D-CTA) (Fig. 1-C) and conventional catheter cerebral angiography (Fig. 2-A, 2-B) showed a 4-mm, round-shaped aneurysm in the right distal branch of the LSA. The right internal carotid artery (ICA) angiography of this patient did not show a typical moyamoya vascular pattern, but the right anterior cerebral artery (ACA) looked stenotic, and there was some collateral flow from the normal middle cerebral artery (MCA) (Fig. 2-A). In addition, his left cervical ICA was obstructed, and the left branch of the ICA showed probable moyamoya disease (Fig. 3-A, 3-B).
We performed a pterional craniotomy for aneurysm clipping five days after the initial attack. We identified and evacuated the right basal ganglia hematoma, and then we performed proximal clipping of the parent artery of the aneurysm (Fig. 4-A, 4-B). We incised the aneurysm, removed the partially thrombosed contents and then we wrapped the aneurysm with cotton and fibrin glue. One day later, the patient suffered from postoperative bleeding around the operation site as well as increased IVH, but 3D-CTA showed good proximal clipping and the aneurysm had disappeared. The patient’s mentality was aggravated after the recurrent bleeding and we performed external ventricular drainage (EVD) on the first postoperative day. The EVD was maintained and drainage was performed for seven days and then the drainage tube was removed. After the removal of the EVD, hydrocephalus gradually developed, so we performed lumbar CSF drainage twice, and then we implanted a ventriculo-peritoneal (V-P) shunt. The patient did well after the final shunt surgery and rehabilitation. He was discharged about two months after admission with minor cognitive dysfunction. However, a recurrent ICH was noted at the left basal ganglia about one year later (Fig. 5-A, 5-B). Repeated cerebral angiography showed that the previous right distal LSA aneurysm had disappeared, and there was no newly developed aneurysm (Fig. 6-A, 6-B). The recurrent ICH did not seem to be related to the previous aneurysm; it might have been due to the rupture of the previously noted left moyamoya-like vessels. The amount of recurrent ICH was small, so we administered conservative treatment. The patient presently has mild cognitive dysfunction, but he has no motor weakness or significant neurologic deficit. We continue to conduct regular follow-up and close neurological observation.
Discussion
On our review of the literature, we found about 25 cases of distal LSA aneurysms have been reported since 1980 (Table 1). In Korea, Han et al.7) reported the first case of ruptured distal LSA aneurysm associated with ipsilateral middle cerebral artery (MCA) occlusion in 2006. Ahn et al.1) then documented two cases of distal LSA aneurysms in 2007, including a case of Han et al.7) Although the number of cases is small, these aneurysms showed some characteristic features, such as young age of onset, no female predominance, usually non-hypertensive with deep location and many associated vascular lesions.1) In the literature, the age of the patients has widely varied from 2 months to 69 years;10)18) the number of male and female patients was similar. Most of these aneurysms originated from the lateral LSA territories and this results in basal ganglia hemorrhage. The aneurysms were usually small (<5mm) and in a deep location. The aneurysm in our case was also small, 4mm and it was located in the deep basal ganglia. Ahn et al.1) classified five types of hemorrhage in the reported cases as 1) isolated ICH, 2) isolated IVH, 3) ICH and IVH, 4) ICH and SAH and 5) a combination of all three types. ICH and IVH are the most common types of hemorrhage of distal LSA aneurysms including our case.3)7)10)14)16)17-19)22) There are many possible underlying diseases in these types of aneurysms, including moyamoya disease or moyamoya-like disease, arteriovenous malformation (AVM), systemic lupus erythematosus (SLE), brain tumor, vasculitis, trauma, arteriosclerosis and hypertension. The associated diseases of the reported cases were moyamoya disease,2)4)5)7)8)12)13)17)21)23)24) AVM,19) SLE11) and brain tumor.25) Moyamoya disease and moyamoya-like disease are the most common types of underlying disease, including in this case, but a history of hypertension was rarely noted.4)15)20) Moyamoya disease is often accompanied by cerebral aneurysms and its incidence has been reported in 3~14% of the cases.2)12) Our patient presented with occlusion of the left ICA; therefore, the left ICA branch had developed probable moyamoya disease. Although the right ICA angiography of this patient did not show a typical moyamoya vascular pattern, the right ACA looked stenotic, and there was some collateral flow from the normal MCA. In addition, the aneurysm and many of the surrounding small vessels seemed fragile in the operation field, and they were similar to moyamoya vessels. Therefore, we think the initial ICH with IVH and the recurrent ICH of this case were due to the rupture of a moyamoya-related aneurysm and the fragile moyamoya-like vessels. Moyamoya-related aneurysms are usually located in the basal ganglia or periventricular white matter.13) Moyamoya disease usually presents with hemorrhage, which due to a rupture of the true aneurysm or pseudoaneurysm, or fragile moyamoya vessels. Although moyamoya-related aneurysms are often regarded as pseudoaneurysms and they can spontaneously regress on the follow-up angiography, they can rerupture (there is a 20~30% incidence of this), which usually results in a poor outcome.12) For this reason, surgical management should be performed in cases with recurrent bleeding or an increasing size of the aneurysm.
The most ideal treatment of LSA aneurysms still remains controversial due to the small number of cases and the undetermined natural course. A few cases were treated conservatively in the literature we reviewed,1)5)6)17)20)21) and two cases were treated with endovascular embolization.8)14) The other cases were managed surgically,3)4)7)9-11)15)18-20)22)23)25) including our case. Conservative treatment and regular follow-up cerebral angiography are adequate management for some lesions such as pseudoaneurysms. However, direct surgery should be considered for typical saccular aneurysms to prevent rebleeding. Surgical treatment is definitely indicated for a growing aneurysm and recurrent intracranial bleeding. The surgical methods include resection of the aneurysm,4)7)10)11)15)16)19) neck clipping4)19)22)23) and proximal clipping.3)4)18) However, one case underwent only hematoma removal.20) Surgical treatments of LSA aneurysms usually have a risk of damage to the eloquent area of the brain due to the deep location of LSA aneurysms in the basal ganglia. Surgical treatments of LSA aneurysms also carry the risk of parent artery occlusion, and this can lead to infarction of the perforating artery and contralateral hemiparesis. Although occlusion of the parent artery may result in neurological deterioration, this procedure may be tolerated when performed in the distal segment because of the rich anastomotic network. Many authors have trapped the parent artery and resected these aneurysms without further neurological damage; however, the parent artery should be preserved, if possible. Gandhi et al.4) reviewed their surgical cases and they insisted that surgical treatment of ruptured LSA aneurysms can be an appropriate, effective and safe therapy. They categorized the morphology of LSA aneurysms into two types based on whether the LSA was not incorporated into the aneurysm dome (Type I) or it was incorporated into the dome (Type II). They were able to spare the parent LSA by performing only aneurysm clipping in three cases of Type I aneurysms. Our case was a Type II aneurysm, so we performed a proximal clipping of the parent artery. It is also important that the underlying structural vascular abnormalities such as AVMs and aneurysms should be appropriately treated to prevent rebleeding. It is difficult to find the parent artery and small aneurysms because of their deep ICH location, so the use of a navigation system or frameless stereotactic guidance may be very useful for localizing and detecting these aneurysms.
Pathologic examinations of these aneurysms were performed in eight of the previous surgical cases, and all of the lesions were revealed to be true aneurysms.6)9)10)14)15)16)18)23) Our case looked like a true aneurysm, but we could not perform a pathologic exam as it was difficult to excise the lesion due to the many fragile surrounding small vessels.
The clinical outcomes of the reported cases have varied, and they mainly depended on the initial neurological status. The outcomes were a good recovery in about half the reported cases,1)3)4)6)7)8)9)16)18)20)21) including this one, moderate to severe disability in some cases,5)10)14)15)19)22)23)25) and death in three cases.1)11)17) The reported cases of distal lenticulostriate artery aneurysms in our literature review are summarized in Table 1.
It has been reported in the literature that spontaneous ICH is associated with a higher incidence of vascular anomalies in young patients who are without preexisting hypertension. Zhu et al.26) suggested that diagnostic cerebral angiography should be considered for all spontaneous ICH patients, except those over 45 years old, with preexisting hypertension in thalamic, putaminal or posterior fossa hemorrhage. Therefore, aggressive investigation in patients who present with spontaneous intracranial hemorrhage should include 3D-CTA, magnetic resonance imaging (MRI) with magnetic resonance angiography (MRA) and conventional cerebral angiography, and especially in those patients who are young or who have no known risk factors such as hypertension, old age or coagulopathy.
Conclusions
Although spontaneous ICH is relatively common, an underlying vascular lesion is rarely found. It might be that most spontaneous ICHs are related to hypertensive ICH, moyamoya disease, AVM, amyloid angiopathy or other vascular anomalies. We think the initial ICH with IVH and the recurrent ICH of this case were due to the rupture of a moyamoya-related aneurysm and the fragile moyamoya-like vessels. When spontaneous ICH occurs in an unusual site, a thorough clinical workup is important to rule out the presence of a structural vascular abnormality. In addition, selection of an appropriate treatment method is mandatory.
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