An unusual ischaemic presentation of thrombosed intracranial mycotic aneurysm with subsequent subarachnoid haemorrhage

  1. Patrick Brown 1 and
  2. Daniel Fulks 2
  1. 1 Radiology and Neurological Surgery, Wake Forest Baptist Medical Center, Winston-Salem, North Carolina, USA
  2. 2 Radiology, The University of Tennessee Medical Center, Knoxville, Tennessee, USA
  1. Correspondence to Dr Patrick Brown; patrick.a.brown@wakehealth.edu

Publication history

Accepted:06 Sep 2020
First published:10 Oct 2020
Online issue publication:10 Oct 2020

Case reports

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Abstract

We present an unusual case of acute ischaemic stroke secondary to thrombosed mycotic aneurysm with subsequent early aneurysmal rupture and subarachnoid haemorrhage, successfully treated with endovascular coil embolisation of the thrombosed segment. Imaging correlates are presented demonstrating successful endovascular management despite vessel occlusion precluding angiographic visualisation of the aneurysmal segment. Imaging and clinical follow-up is provided demonstrating durable occlusion and excellent clinical outcome with full functional recovery.

Background

Mycotic aneurysms are an unusual cause of subarachnoid haemorrhage but represent a potentially growing segment of haemorrhagic cerebrovascular disease as the opioid epidemic continues in the USA, with associated increased rates of bacterial endocarditis. Mycotic aneurysms may have varying presentations, including haemorrhagic, secondary to rupture, as well as ischaemic, secondary to thrombosis. Their endovascular management may be challenging due to frequent distal location, lesion friability, as well as occasional occult nature on angiographic studies. While the majority of these types of lesions currently develop in the setting of bacterial endocarditis often related to intravenous drug abuse, it is important to consider other causative factors such as dental or periodontal disease as well as metastatic aneurysm formation in diseases such as cardiac myxomas.

Case presentation

A late middle-aged male patient presented to the emergency department with the acute onset of left-sided facial and hemibody weakness, sensory neglect and dysarthria with National Institutes of Health (NIH) stroke scale of 9. He underwent CT and CT angiography demonstrating branch occlusion within the right M2 superior division with hypoattenuation of the associated vascular territory. A small rounded focus of associated hyperattenuation was noted distal to the site of vessel occlusion, initially interpreted as focal haemorrhagic transformation (figure 1). Given the concern for focal haemorrhagic transformation, the patient was not deemed a candidate for mechanical thrombectomy or intravenous thrombolytics.

Figure 1

(A) Axial non-contrast CT head demonstrating rounded hyperattenuating focus within the right Sylvian fissure (arrow) and (B) developing right middle cerebral artery (MCA) infarction (*).

The patient was transferred to the intensive care unit, where he subsequently developed a severe headache and decreased level of consciousness, Glasgow Coma Scale 13, with unchanged pattern of focal neurologic deficits. He was taken for follow-up CT which demonstrated diffuse sulcal and cisternal subarachnoid haemorrhage, centred predominantly within the right Sylvian fissure. CT angiography was repeated which demonstrated persistent occlusion of the right M2 superior division branch and no demonstrable source of subarachnoid haemorrhage (figure 2).

Figure 2

(A) Follow-up non-contrast CT after exam decline demonstrating new cisternal subarachnoid haemorrhage (*) and (B) CT angiogram demonstrating non-filling of right M2/M3 branch (arrow).

Neurointerventional service was consulted, with plan for angiographic evaluation and possible endovascular management. During preoperative assessment, the patient was noted to have profound dental and periodontal disease and endorsed self-extracting an abscessed tooth approximately 2 weeks prior to presentation. The patient and family denied any history of intravenous drug abuse. Toxicology screen was negative.

He was taken for angiography which demonstrated branch occlusion within the right M2 superior division and no angiographic demonstration of aneurysm or other haemorrhagic source. Given the provided history, underlying thrombosed mycotic aneurysm was considered to be the most likely differential consideration, and endovascular coil embolisation was performed at the level of the occlusion (figure 3). Postoperative CT head demonstrated localisation of the coil mass at the site of the hyperattenuating focus noted on the presentation head CT, suggestive of successful coil embolisation of angiographically occult mycotic aneurysm. Three-month follow-up CT head demonstrated well-developed infarction limited to the region of hypoattenuation seen on initial presentation (figure 4).

Figure 3

(A,B) Anteroposterior (AP) and lateral right internal carotid angiogram demonstrating right M2/M3 branch occlusion (blue arrow) with no aneurysmal opacification and (C,D) subsequent coil mass placement (red arrows).

Figure 4

(A) Postoperative axial non-contrast CT head demonstrating embolic coil placement at the location of initial hyperdensity (arrow) and (B) evolved right MCA territory infarct (*) in area of early ischaemia seen in figure 1.

Differential diagnosis

Differential considerations for this case initially included haemorrhagic transformation of ischaemic stroke, thrombosed middle cerebral artery (MCA) aneurysm and thrombosed MCA mycotic pseudoaneurysm. The initial imaging with rounded hyperattenuating structure within the right Sylvian fissure represented an unusual morphology and location for haemorrhagic transformation, and as such early haemorrhagic transformation presenting in this manner would be considered very atypical. For these reasons, the alternative differential considerations were thought to be more likely. Cerebral aneurysms, particularly when thrombosed, may present with focal rounded hyperattenuation with Hounsfield units similar to that of acute blood products (approximately 60–90 HU). The subsequent development of cisternal subarachnoid haemorrhage further supported the diagnosis of thrombosed aneurysm or pseudoaneurysm. Given the history of recent dental abscess with self-performed tooth extraction, mycotic aneurysm became the leading differential consideration.

Treatment

Cerebral angiography was performed under general anaesthesia. A six French introducer sheath was placed in the right common femoral artery, and a five French angled tapered GlideCath (Terumo, Tokyo, Japan) was utilised to perform full diagnostic cerebral angiography, demonstrating occlusion of right M2 superior division branch and no other identifiable source of haemorrhage. Subsequently, an arrangement of a NavienTM (ev3, Plymouth, MN) 0.072″ support catheter with coaxial five French Sofia (Microvention, Aliso Viejo, California, USA) intermediate catheter and Headway Duo (Microvention) microcatheter was advanced over a Synchro-2 (Stryker, Kalamazoo, Michigan, USA) 0.014″ microwire. The occluded branch was accessed with the microcatheter and gentle microangiography was performed confirming branch occlusion. The microcatheter was distally navigated within the branch, and coil embolisation was carried out with Target (Stryker) detachable coils. Follow-up angiography was performed demonstrating occlusion of the segment and stable positioning of the coil mass with no contrast opacification distal to the coil mass.

Outcome and follow-up

The patient was transferred to the intensive care unit, where he recovered well with subsequent improvement in his stroke symptoms. Follow-up angiography was performed during his hospital stay demonstrating stable coil mass positioning with durable occlusion. He was discharged to home after recovery.

At 3-month clinic follow-up, the patient had resolution of stroke symptoms and was neurologically intact with NIH stroke scale 0. He had returned to work, and dental consult was obtained for further tooth extraction.

Discussion

Cerebral mycotic aneurysms are a rare cause of intracranial haemorrhage and represent only 0.7% to 5.4% of all cerebral aneurysms.1 Their pathogenesis involves invasion and inflammatory destruction of vessel wall secondary to septic emboli with subsequent vascular dilatation and aneurysmal formation.2 By nature, these lesions are extremely friable due to their incomplete vascular wall and functionally behave as pseudoaneurysms. The presence of cerebral mycotic aneurysm in the setting of infective endocarditis carries a mortality rate of up to 13%, with mortality rates approaching 80% in the setting of rupture.3 The most common causes of infective bacteraemia are intravenous drug abuse and poor dental hygiene. Cerebral mycotic aneurysms may also occur by direct invasion from adjacent infectious processes such as bacterial meningitis.4

When a cerebral mycotic aneurysm is suspected, vascular imaging is indicated, with the gold standard evaluation being catheter angiography.5 Cerebral mycotic aneurysms tend to be peripherally located with fusiform morphology, though can occasionally be proximal in location with features similar to those of berry aneurysms. In these cases, adjacent arterial stenoses or occlusions, rapid change in size or morphology or multiplicity of aneurysms may favour a diagnosis of mycotic aneurysm.6 Ruptured cerebral mycotic aneurysms should be immediately secured if possible, either by endovascular or surgical means, while unruptured aneurysms may undergo a trial of antibiotic therapy versus procedurally securing the aneurysm. However, if subsequent imaging demonstrates progressive growth, procedural management is indicated.4 Cerebral mycotic aneurysms may demonstrate a quite dynamic angiographic appearance and may be angiographically occult on initial imaging, becoming visible on subsequent examinations.7 For these reasons, if there is high clinical suspicion for cerebral mycotic aneurysm with a negative initial evaluation, early repeat angiography should be considered. However, the presence of a ruptured angiographically occult cerebral mycotic aneurysm presents a much more challenging scenario.

In this case study, the presence and location of a putative ruptured mycotic aneurysm can be inferred from the presentation non-angiographic CT head. Despite the inability to visualise the lesion with angiographic techniques, decision was made to proceed with endovascular coil embolisation/vessel sacrifice at the site of vessel occlusion as demonstrated on angiography. While successful and safe embolisation procedures typically require adequate visualisation of the target lesion, in this case, conservative measures and/or delayed imaging were considered of greater risk for further haemorrhagic complications than potential for ischaemia related to non-target embolisation. The haemorrhagic source was inferred from the presentation CT head and subsequently confirmed on post-procedure imaging. In our case there were no ischaemic or haemorrhagic complications, and the patient made a full neurologic recovery. We suggest from our experience that aggressive measures are indicated when managing ruptured cerebral mycotic aneurysms, even in the setting of angiographically occult lesions.

Learning points

  • Ruptured cerebral mycotic aneurysms carry an extremely high mortality rate, approaching 80%.

  • The most common current clinical scenario is bacteraemia related to intravenous drug abuse, but other sources such as dental infection should also raise suspicion.

  • Cerebral mycotic aneurysms are frequently angiographically occult, and other non-angiographic modalities may be useful in demonstrating their presence and location.

  • Aggressive procedural management of ruptured cerebral mycotic aneurysms is indicated due to their high mortality rates.

  • Successful procedural management of angiographically occult ruptured cerebral mycotic aneurysms may be aided by integration of findings of other non-angiographic studies.

Footnotes

  • Twitter @ptrxbrn

  • Contributors PB helped in writing manuscript text, figure editing and caption editing. DF helped with the figures.

  • 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.

References

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