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Case report
Ruptured lenticulostriate artery aneurysm: a report of a case treated with endovascular embolisation
  1. Luca Roccatagliata1,2,
  2. Marco Pileggi3,
  3. Alessandro Cianfoni3,4 and
  4. Jan Gralla4
  1. 1Department of Health Sciences (DISSAL), University of Genoa, Genoa, Liguria, Italy
  2. 2Department of Neuroradiology, Ospedale Policlinico San Martino IRCCS, Genoa, Liguria, Italy
  3. 3Department of Neuroradiology, Neurocenter of Southern Switzerland, Lugano, Switzerland
  4. 4Department of Diagnostic and Interventional Neuroradiology, Inselspital University Hospital Bern, Bern, Switzerland
  1. Correspondence to Dr Alessandro Cianfoni; alessandro.cianfoni{at}eoc.ch

Abstract

A 65-year-old woman presented to the emergency department with sudden onset of left-sided weakness, headache and vomiting. A cerebral CT showed an acute intracerebral haemorrhage involving the right caudate nucleus and lentiform nucleus with mild midline shift and intraventricular extension. CT angiography did not reveal aneurysm or other vascular anomaly. Conventional cerebral angiography demonstrated a 3 mm right medial lenticulostriate branch aneurysm, arising from the right anterior cerebral artery (ACA). Endovascular treatment was performed from the left internal carotid via the anterior communicating artery into the right ACA. Complete occlusion was achieved with injection of N-butyl-2-cyanoacrylate. The patient had neurological rehabilitation during hospitalisation followed by outpatient physical therapy. Two years later, clinical follow-up demonstrated excellent recovery.

  • interventional radiology
  • neuroimaging

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Background

Lenticulostriate artery aneurysms have been reported as a very rare cause of intracerebral haemorrhage (ICH) at the level of the basal ganglia.1 2 Patients with ruptured aneurysm of a lenticulostriate artery have been managed conservatively or rarely treated with open surgery, endovascular approaches or gamma-knife radiosurgery, but it is uncertain which option should be preferred.

We report a case of a woman with a ICH caused by a rupture of aneurysm in a lenticulostriate artery, successfully treated by endovascular embolisation despite a challenging microcatheter navigation.

In patients with ICH in the basal ganglia, lenticulostriate artery aneurysms should be actively sought; in selected cases, endovascular treatment can be effective in protecting from rebleeding.

Case presentation

A 65-year-old woman presented to the emergency department with sudden onset of left-sided weakness, headache and vomiting. At admission, blood pressure was 160/105 mm Hg. She had a right gaze deviation and left hemiparesis. Glasgow coma scale score was 12. Coagulation test and platelets count were within normal limits; usual medications included aspirin but not anticoagulants. Her baseline pressure at a recent outpatient clinic evaluation was 145/90 mm Hg.

Investigations

A cerebral CT showed an acute ICH measuring approximately 18 mL in volume involving the right caudate nucleus and lentiform nucleus with mild midline shift and effacement of the right lateral ventricle. There was intraventricular extension of haemorrhage with signs of early hydrocephalus. (figure 1A).

Figure 1

(A) Unenhanced CT demonstrates intraparenchymal haematoma at the level of the basal ganglia in association with intraventricular haemorrhage. (B) Angiography demonstrates a 3 mm aneurysm (black arrow) along a right medial lenticulostriate branch arising from the anterior cerebral artery. (C) Pre-embolisation superselective microcatheter injection of the lenticulostriate branch harbouring the aneurysm from ipsilateral carotid, demonstrating recurrent origin of the target vessel. (D) Endovascular embolisation with NBCA performed from controlateral internal carotid through the anterior communicating artery. (E) Post-treatment unenhanced CT demonstrates NBCA cast (black arrow) and (F) post-treatment angiographic control demonstrates complete occlusion of the aneurysm. NBCA, N-butyl-2-cyanoacrylate.

CT angiography (CTA) did not reveal aneurysm or other vascular anomaly. A ventricular drainage was positioned. Conventional cerebral angiography was performed 36 hours later and demonstrated a 3 mm right medial lenticulostriate branch aneurysm, arising from the right anterior cerebral artery (ACA) (figure 1B).

Treatment

Endovascular treatment was performed under general anaesthesia. As the lenticulostriate perforator was inaccessible from the ipsilateral carotid, a 1.2F microcatheter (Sonic, Balt/France) with a 0.007 inch guidewire (Hybrid, Balt/France) was advanced from the left internal carotid via the anterior communicating artery into the right ACA and the origin of the target vessel (figure 1C–D). Complete occlusion was achieved with injection of N-butyl-2-cyanoacrylate (NBCA), in a mixture with ethiodized oil (Lipiodol), 1:6. Final angiogram proved occlusion (figure 1E–F).

Outcome and follow-up

The patient had neurological rehabilitation during hospitalisation followed by outpatient physical therapy. Two years later, clinical follow-up demonstrated excellent recovery with only a mild memory impairment.

Discussion

This patient presented with an ICH caused by the rupture of a lenticulostriate artery aneurysm. These aneurysms are rare, with less than 60 cases reported in the literature.1–3 They are typically small, with an average maximum diameter of 3.1 mm (range 1–6 mm). Different conditions have been described as related with lenticulostriate arteries aneurysms among which moya-moya (reported in 15 cases) and systemic hypertension (in 12 cases). Isolated cases have also been reported in association with vasculitis, fibromuscular dysplasia, systemic lupus erythematous, cocaine use and with the presence of brain arteriovenous malformations.

The rupture of a lenticulostriate artery aneurysm typically causes an intraparenchymal haematoma at the level of the basal ganglia, often in association with intraventricular haemorrhage while the presentation with subarachnoid haemorrhage is less common. Timely diagnosis of these lesions can be difficult; the basal ganglia location is common in ICH secondary to hypertension, thus potentially leading to a misdiagnosis of hypertension associated ICH without suspecting an underlying vascular lesion. CTA is routinely acquired in intracranial bleedings diagnostic work-up; CTA has a sensitivity for small aneurysms, less than 3 mm, of approximately 90%.4 As a matter of fact, in our patient, CTA did not reveal the aneurysm, which was subsequently identified at conventional angiography. Conventional angiography is usually considered in young patients and in non-hypertensive patients when CTA fails to identify a vascular cause for haemorrhage. Interestingly, even in patients with systemic hypertension, a structural cause for ICH can be identified. In a study on hypertensive subjects with ICH, autopsy revealed a vascular structural lesion, either an aneurysm or an arteriovenous malformation, in 35 out of 144 patients.5 From the neuropathological point of view, lenticulostriate artery aneurysms are categorised among dissecting microaneurysms by some authors.6 It could be considered that pathological substrate might vary according to the underlying associated condition (ie, Moya-moya versus hypertension). Takebayashi and Kaneko7 in a pathological study of 11 brains with hypertensive ICH found two ‘miliary aneurysms’ of the lenticulostriate arteries and described in the arterial wall connected to the aneurysm severe degeneration of smooth muscle cells with fibrin and plasma exudates in the subendothelium; the adjacent branches of the lenticulostriate arteries demonstrated severe atherosclerosis, segmental dilatation with or without disruption of the elastic lamina and lipid laden macrophages in the intima. The natural history of intracranial haemorrhage associated with lenticulostriate aneurysms is not well known due to the low number of described cases. It should be noted that the well-established data on the high risk of repeated aneurismal rupture in the first days after subarachnoid aneurismal haemorrhage caused by the rupture of saccular aneurysms are not directly applicable in the case of intraparenchymal cerebral haematomas caused by the rupture of lenticulostriate aneurysms. Conservative management has been advocated by some authors,2 at least when there is neither haematoma expansion nor increase in size of the aneurysm at close angiographic follow-up. Treatment options include open surgery or endovascular approaches;2 3 8 gamma-knife radiosurgery has also been proposed.9 One advantage of open surgery is that it enables the evacuation of the intraparenchymal haematoma but the deep location of the aneurysms and their small size makes surgery challenging. Gandhi and coauthors8 classified lenticulostriate aneurysms in type I and type II. In type I aneurysms, the dome of the lesion is not incorporated into the lenticulostriate artery and these aneurysms can be treated by surgical clipping safeguarding the patency of the parent artery. In type II aneurysms, the aneurismal dome is incorporated into the lenticulostriate artery, requiring trapping, excision or lenticulostriate artery sacrifice with proximal occlusion of the vessel. More recently, endovascular approaches have been proposed for the treatment of these lesions; difficulties associated with endovascular treatment include the complexity in accessing the aneurysm with the microcatheter navigating extremely tortuous and small calibre arteries. NBCA has been used as embolic material, more frequently to occlude the parent vessel together with the aneurysm as in our case. Tsai and coauthors10 report embolisation with NBCA with preservation of the feeding artery. Onyx has also been used as embolic material. In one patient, gamma knife radiosurgery has been used to treat a ruptured distal lenticulostriate arteries aneurysm; obliteration of the lesion was confirmed 22 months after treatment.9 The delay between radiation treatment and obliteration might not be efficient in protecting from rebleeding. In our case of a patients presenting with ICH due to a ruptured lenticulostriate aneurysm, endovascular embolisation was technically feasible and was performed with a good clinical result.

Learning points

  • Endovascular embolisation can be technically feasible with excellent clinical results in patients with intracerebral haemorrhage due to a ruptured lenticulostriate aneurysm.

  • N-butyl-2-cyanoacrylate has been used as embolic material, more frequently to occlude the parent vessel together with the aneurysm as in our case.

  • Uncertainties persist on the best management of ruptured lenticulostriate aneurysms.

References

Footnotes

  • Contributors LR: review of clinical and radiological data, manuscript writing, review of literature, manuscript review and editing. MP: manuscript writing, manuscript review and editing. AC: review of clinical and radiological data, manuscript writing, manuscript review and editing. JG: interpretation and review of radiological data, manuscript writing, manuscript review and editing.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent for publication Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.