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KOR J CEREBROVASCULAR SURGERY September 2011 Vol. 13 No 3, page 129-136 Distal Lenticulostriate Artery Aneurysm Presenting With Spontaneous Intracerebral and Intraventricular Hemorrhage : A Case Report and a Review of the Literature Department of Neurosurgery, Chungnam National University School of Medicine, Daejeon, Korea Jae-Kyung Sung, M.D.・Hyeon-Song Koh, M.D.・Chang-Woo Kang, M.D.・Hyon-Jo Kwon, M.D. Jin-Young Youm, M.D.・Seon-Hwan Kim, M.D. ABSTRACT 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-dimen- sional computed tomography angiography (3D-CTA) and conventional cerebral angiography showed a 4 mm, round-shaped aneur- ysm in the right distal LSA and this was combined with moyamoya-like disease. We performed proximal clipping of the aneur- ysm 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. (Kor J Cerebrovascular Surgery 13(3):129-136, 2011) KEY WORDS : Aneurysm・Distal lenticulostriate artery・Hemorrhage ports of distal LSA aneurysms in the medical literature. Introduction Due to its rarity, we report here a case of a ruptured dis- tal LSA aneurysm that presented as spontaneous ICH and Spontaneous intracerebral hemorrhage (ICH) is reported intraventricular hemorrhage (IVH) in a healthy male pa- in about 10~20% of all strokes,4)7) and it frequently caus- tient, and we review the relevant literature. es significant neurologic deficits. Many of these hemor- rhages are related to microaneurysms that form in pa- Description of the case tients with hypertension and they are rarely discovered by cerebral angiography. Aneurysms arising from the branch- A 66-year-old man was transferred from a local clinic es of the distal lenticulostriate artery (LSA) are a rare with a continuous intractable headache that had started cause of spontaneous ICH; there are about 25 case re- three days previously. He had no history of hypertension, diabetes or taking any specific medication and he had no 논문접수일 : 2011년 5월 18일 definite neurologic deficit. A brain computed tomography 심사완료일 : 2011년 8월 8일 (CT) scan showed a right basal ganglia ICH and IVH 교신저자 : Hyeon-Song Koh, M.D., Ph.D., Department of Neurosurgery, Chungnam National University School of Medicine, 33 Munhwa-ro, (Fig. 1-A, 1-B). Three-dimensional CT angiography (3D-CTA) Jung-gu, Daejeon 301-721, Korea (Fig. 1-C) and conventional catheter cerebral angiography Tel : (042) 280-7369 ∙ Fax : (042) 280-7363 (Fig. 2-A, 2-B) showed a 4-mm, round-shaped aneurysm Email : [email protected] 129 Distal LSA Aneurysm A B C Fig. 1. (A)The brain computed tomography (CT) scan at admission shows intracerebral hemorrhage on the right basal gan- glia and (B) intraventricular hemorrhage. (C) Three-dimensional CT angiography (3D-CTA) shows a small, round-shaped aneurysm (arrow) on the branch of the right distal lenticulostriate artery. A B Fig. 2. (A, B) Catheter cerebral angiography in the right internal carotid artery shows a small, round-shaped aneurysm (arrow) on the right distal lenticulostriate artery. in the right distal branch of the LSA. The right internal clipping five days after the initial attack. We identified carotid artery (ICA) angiography of this patient did not and evacuated the right basal ganglia hematoma, and then show a typical moyamoya vascular pattern, but the right we performed proximal clipping of the parent artery of anterior cerebral artery (ACA) looked stenotic, and there the aneurysm (Fig. 4-A, 4-B). We incised the aneurysm, was some collateral flow from the normal middle cerebral removed the partially thrombosed contents and then we artery (MCA) (Fig. 2-A). In addition, his left cervical wrapped the aneurysm with cotton and fibrin glue. One ICA was obstructed, and the left branch of the ICA day later, the patient suffered from postoperative bleeding showed probable moyamoya disease (Fig. 3-A, 3-B). around the operation site as well as increased IVH, but We performed a pterional craniotomy for aneurysm 3D-CTA showed good proximal clipping and the aneur- 130 Kor J Cerebrovascular Surgery 13(3):129-136, 2011 Jae-Kyung Sung・Hyeon-Song Koh・Chang-Woo Kang・Hyon-Jo Kwon・Jin-Young Youm・Seon-Hwan Kim A B Fig. 3. (A, B) Left cerebral angiography shows total occlusion of the left cervical internal carotid artery (arrow), and this suggested of probable moyamoya disease. A B Fig. 4. (A) The intraoperative findings show the round-shaped aneurysm on the right distal lenticulostriate artery (B) and proximal clipping of the aneurysm. ysm had disappeared. The patient’s mentality was ag- was removed. After the removal of the EVD, hydro- gravated after the recurrent bleeding and we performed cephalus gradually developed, so we performed lumbar external ventricular drainage (EVD) on the first post- CSF drainage twice, and then we implanted a ven- operative day. The EVD was maintained and drainage triculo-peritoneal (V-P) shunt. The patient did well after was performed for seven days and then the drainage tube the final shunt surgery and rehabilitation. He was dis- Kor J Cerebrovascular Surgery 13(3):129-136, 2011 131 Distal LSA Aneurysm A B Fig. 5. (A, B)The computed tomography scan one year after the initial attack shows another intracerebral hemorrhage (ICH) on the left basal ganglia. Also shown are an aneurysm clip on the right basal ganglia and a ventricular shunt catheter. A B Fig. 6. (A) Repeated cerebral angiography one year later shows the disappearance of the previous aneurysm on the right distal lenticulostriate artery. (B) There is no de novo aneurysm seen on the left cerebral angiogram. charged about two months after admission with minor there was no newly developed aneurysm (Fig. 6-A, 6-B). cognitive dysfunction. However, a recurrent ICH was not- The recurrent ICH did not seem to be related to the pre- ed at the left basal ganglia about one year later (Fig. vious aneurysm; it might have been due to the rupture of 5-A, 5-B). Repeated cerebral angiography showed that the the previously noted left moyamoya-like vessels. The previous right distal LSA aneurysm had disappeared, and amount of recurrent ICH was small, so we administered 132 Kor J Cerebrovascular Surgery 13(3):129-136, 2011 Jae-Kyung Sung・Hyeon-Song Koh・Chang-Woo Kang・Hyon-Jo Kwon・Jin-Young Youm・Seon-Hwan Kim conservative treatment. The patient presently has mild fore, the left ICA branch had developed probable moya- cognitive dysfunction, but he has no motor weakness or moya disease. Although the right ICA angiography of significant neurologic deficit. We continue to conduct reg- this patient did not show a typical moyamoya vascular ular follow-up and close neurological observation. pattern, the right ACA looked stenotic, and there was some collateral flow from the normal MCA. In addition, Discussion the aneurysm and many of the surrounding small vessels seemed fragile in the operation field, and they were sim- On our review of the literature, we found about 25 ilar to moyamoya vessels. Therefore, we think the initial cases of distal LSA aneurysms have been reported since ICH with IVH and the recurrent ICH of this case were 1980 (Table 1). In Korea, Han et al.7) reported the first due to the rupture of a moyamoya-related aneurysm and case of ruptured distal LSA aneurysm associated with ip- the fragile moyamoya-like vessels. Moyamoya-related aneur- silateral middle cerebral artery (MCA) occlusion in 2006. ysms are usually located in the basal ganglia or periven- Ahn et al.1) then documented two cases of distal LSA tricular white matter.13) Moyamoya disease usually pres- aneurysms in 2007, including a case of Han et al.7) ents with hemorrhage, which due to a rupture of the true Although the number of cases is small, these aneurysms aneurysm or pseudoaneurysm, or fragile moyamoya showed some characteristic features, such as young age of vessels. Although moyamoya-related aneurysms are often onset, no female predominance, usually non-hypertensive regarded as pseudoaneurysms and they can spontaneously with deep location and many associated vascular lesions.1) regress on the follow-up angiography, they can rerupture In the literature, the age of the patients has widely varied (there is a 20~30% incidence of this), which usually re- from 2 months to 69 years;10)18) the number of male and sults in a poor outcome.12) For this reason, surgical man- female patients was similar. Most of these aneurysms or- agement should be performed in cases with recurrent iginated from the lateral LSA territories and this results bleeding or an increasing size of the aneurysm. in basal ganglia hemorrhage. The aneurysms were usually The most ideal treatment of LSA aneurysms still re- small (< 5 mm) and in a deep location. The aneurysm in mains controversial due to the small number of cases and our case was also small, 4 mm and it was located in the the undetermined natural course. A few cases were treated deep basal ganglia. Ahn et al.1) classified five types of conservatively in the literature we reviewed,1)5)6)17)20)21) and hemorrhage in the reported cases as 1) isolated ICH, 2) two cases were treated with endovascular embolization.8)14) isolated IVH, 3) ICH and IVH, 4) ICH and SAH and 5) The other cases were managed surgically,3)4)7)9-11)15)18-20)22)23)25) a combination of all three types.

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