Netherlands Journal of Critical Care

NETH J CRIT CARE - VOLUME 14 - NO 1 - FEBRUARY 201032

CASE REPORT

MB Aerts1, YDI Vandevivere1, GN Beute2, JAH van Oers1

1 Department of Intensive Care St. Elisabeth Hospital, Tilburg, The Netherlands

2 Department of Neurosurgery St. Elisabeth Hospital, Tilburg, The Netherlands

Abstract - We present two cases of subarachnoid haemorrhage (SAH) caused by ruptured infectious intracranial aneurysms secondary

to endocarditis. The first patient presented with neurological signs of an SAH. Fever and the presence of a previously undiagnosed heart

murmur were reasons to obtain blood cultures and initiate further diagnostic workup for endocarditis. The second patient presented

with neurological symptoms during the evaluation and treatment of previously diagnosed endocarditis. Loss of consciousness due to

a ruptured aneurysm on the 27th day of treatment prompted further research. Using these two cases as illustrations, we want to draw

attention to the dangerous neurological complications that can occur during an episode of endocarditis and call for timely evaluation.

Keywords - Subarachnoid haemorrhage, SAH, bacterial endocarditis

A subarachnoid haemorrhage as a complication of bacterial endocarditis

Copyright © 2010, Nederlandse Vereniging voor Intensive Care. All Rights Reserved. Received August 2009; accepted October 2009

Introduction

Ever since the first description of endocarditis by W. Osler,

neurological symptoms have been known to complicate episodes

of endocarditis. The estimated incidence of neurological

symptoms complicating endocarditis is high, between 20 and

40% [1]. Cerebral ischaemia is the most frequently encountered

complication (20%), followed by encephalopathy (9%), meningitis

(7%) and rarely, SAH (1-2%). [1] To date, SAH heralding

endocarditis has been reported only 11 times in the literature

(Table 1). In this case report, we will present two illustrative cases

of SAH, both heralding and complicating bacterial endocarditis.

Case reports

Patient A, a 35-year-old woman was admitted to the emergency

department of a regional hospital with sudden loss of consciousness.

Her initial Glasgow coma score (GCS) was E3M6V4 and she had

left-sided hemiparesis. Her medical history revealed no previous

illnesses or hospital admission. There was no evidence to suggest

intravenous drug abuse. Hetero-anamnesis revealed that she

had been suffering from general malaise and arthralgia for the

past few months. A CT scan (Figure 1 a) showed intracerebral

haemorrhage with blood in the Fissura Sylvii, indicative of SAH.

The patient was referred to our neurosurgical centre for further

diagnosis and therapeutic intervention. On physical examination

at our hospital, her blood pressure was 120/70 mmHg, pulse rate

70 beats per min, and temperature 38.5 oC. Inspection of the

mouth and throat revealed no abnormalities. Her lungs were clear

on auscultation and a new onset systolic heart murmur degree III/

VI, punctum maximum (p.m.) apically radiating to the left axillar

region, was found. Examination of the skin and nails revealed no

stigmata or endocarditis. Neurological examination showed a GCS

of E4M6V4 with a left hemiparesis. Laboratory studies included:

CRP 252 mg/l (0-10 mg/l), WBC 18.0 10e9/l (4.0-10 10e9/l), Hb

6.0 mmol/l (7.5-10mmol/l), and prothrombin time 16.0s (10-13.5s).

All other laboratory studies were within normal range. Blood

cultures obtained at the time of admission revealed Streptococcus

viridans. Transoesophageal echocardiography confirmed the

clinical suspicion of endocarditis. Intravenous benzylpenicillin two

million IU six times a day and tobramycin 320 mg once a day,

were started immediately. Cerebral angiography on admission

(Figure 1b) showed a small aneurysm (2x6 mm) distally on the right

middle cerebral artery (M3 branch). Initially selective coiling of the

aneurysm was attempted. Due to its distal localizationthis was

unsuccessful and coiling of the anterior M2 branch was performed.

During the coiling procedure, vascular vasospasms were observed

and treated with 2 mg nimodipin intravascularly. No further

complications occurred during coiling. Post-coiling, the patient’s

GCS diminished to E1M4V1. She was admitted to our intensive

care unit, where she had to be intubated and mechanical ventilation

was started. A CT scan of her brain showed a haemorrhagic

infarct in the right arteria cerebri media area with compression

and midline shift. An emergency hemicraniectomy was performed

and the haematoma was evacuated. Postoperatively, the patient’s

neurological state improved to a maximal GCS score. Left

hemiparesis with an additional neglect on the left side remained.

On day 3 the patient was successfully detubated and on day 6

she was able to be transferred to the neurosurgical ward . Weekly

echocardiography and intravenous antibiotics were continued for

12 weeks. No further destruction of the mitral valve was detected

on echocardiography. Three months later, the patient was seen at

our outpatient clinic. She had regained good cognitive function.

Left-sided hemiparesis remained. The bone flap was re-implanted

without further complications. Because of this successful

recovery, the aneurysm was left in situ and no further culture of the

aneurysm could be performed to officially establish the diagnosis

of an infectious intracranial aneurysm.

JAH van Oers

E-mail: j.v.oers@elisabeth.nl

Correspondence

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Netherlands Journal of Critical Care

A subarachnoid haemorrhage as a complication of bacterial endocarditis

Patient B, a 61-year-old male, was admitted to a regional

hospital for analysis with symptoms of fever, weight loss and

general malaise. His medical history showed gout, chronic

obstructive and restrictive pulmonary disease and an aortic

valve sclerosis with mild aortic valve insufficiency and diastolic

dysfunction. The patient did not use anticoagulants. There was

no evidence of drug abuse. On physical examination his blood

pressure was 110/70 mmHg, pulse rate 80 beats per min, and

temperature 39.3 oC. Inspection of the mouth and throat revealed

no abnormalities. His lungs were clear on auscultation. Apart

from a pre-existent midsystolic heart murmur degree III/VI, p.m.

radiating apically to the left axillar region, no new heart murmur

was heard. Examination of the skin and nails revealed no stigmata

or endocarditis. Lab studies included BSE 43mm/u (<20mm/u),

CRP 85mg/l (1-10mg/l), Hb 5.4 mmol/l (8.5-11mmol/l), and WBC

9.3 10e9/l (4.0-10 10e9/l). All other lab results were within the

normal range. Blood cultures obtained upon admission showed

Streptococcus sanguinis. Echocardiography showed aortic

valve insufficiency degree III-IV/VI, due to valvular vegetation.

The diagnosis of endocarditis was established, for which he

was treated with intravenous benzylpenicillin two million IU six

times a day and gentamycin 80mg three times a day. On the

27th day of treatment he developed diminished consciousness

and left-sided hemiparesis, which quickly deteriorated to a GCS

of E1M1V1 and dilatation of the right pupil. Further physical

examination was unremarkable. A CT scan (Figure 2.a) was

performed, showing an acute right-sided subdural haematoma,

a large intracerebral haemorrhage around the Fissura Sylvii,

and subarachnoidal bleeding. The patient had to be intubated,

ventilated and transferred to our centre for neurosurgical

intervention. An emergency hemicraniectomy was performed and

an aneurysmal lesion in the distal area of the right a. cerebri media

was clipped. Pathological examination of the specimen obtained

during surgery, showed clotted blood and fibrin. Microbiological

investigation of the aneurysm revealed no micro-organisms.

Estimated loss of blood was three litres. Postoperatively, both

pupils were narrow and light reactive. The patient was admitted

to our ICU for mechanical ventilation and further treatment.

A subarachnoid haemorrhage as a complication of bacterial endocarditis

Table 1. Cases of SAH during an episode of endocarditis

AUTHOr N CASES

ENDOCArDITIS

N SAH

(TOTAl)

SAH WITHOUT

ANEUrYSM

SAH WITH PATHOlOGICAl

PrOVEN INFECTIOUS

ANEUrYSM

SAH AS

PrESENTING

SYMPTOM

PUBlISHING

JOUrNAl

Whipple et al (1951) 1 1 0 * 1 Ann Int Med

Tompsett et al (1967) 74 1 1 * * Arch Int Med

Jones et al (1969) 385 6 3 * * Ann Int Med

Gilroy et al (1973) 2 2 0 * * Neurology

Le Frock et al (1973) 2 1 1 * 1 South Med J.

Bingham et al (1977) 2 1 0 0 0 J. Neurosurg

Pruitt et al (1978) 218 4 0 4 0 Medicine

Vincent et al (1980) 1 1 1 0 1 Neurosurgery

Rappaport et al (1981) 1 1 0 0 1 J Iowa Med Soc

Salgado et al (1987) 150 3 0 0 0 Stroke

Brust et al (1990) 17 7 0 * 0 Ann Neurol

Hart et al (1990) 203 1 0 1 1 Stroke

Powell et al (1997) 1 1 0 0 0 J Reprod Med

Takeda et al (1998) 1 1 0 1 0 Clin Neuropathol.

Roberts et al (1998) 1 1 0 * 0 Br J Neurosurg

Bakshi et al (1999) 12 3 3 0 0 J Neuroimaging

Chukwudelunzu et al (2002) 489 8 6 1 2 Eur J Neurol

Yanagihara et al (2003) 1 1 0 0 0 Int Med

Shimizu et al (2006) 1 1 0 0 0 Intern Med

Peters et al (2006) 1 1 0 1 1 Lancet Infect Dis

Kannoth et al(2007) 12 2 0 0 2 J Neurol Sci

Trivedi et al (2008) 1 1 0 0 0 Int J Obstet Anesth

Chang et al (2008) 1 1 0 0 1 Diagn Microbiol Infect Dis

TOTAAL 1577 50 15 8 11

* missing data

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MB Aerts, YDI Vandevivere, GN Beute, JAH van Oers

Postoperatively sedative medication was discontinued but the

GCS score remained E1M1Vtube. Treatment with vaspopressors

and fl uid resuscitation was initiated for haemodynamic instability

with blood pressure dropping to 80/40 mmHg . The next day his

pupils became wide and non-reactive, corneal refl exes were still

present. A CT scan (Figure 2.b) showed oedema and a massive

infarction of the right hemisphere. Unfortunately, during the 48

hours after hemicraniectomy, the patient’s neurological condition

deteriorated. His corneal refl exes diminished and he developed

diabetes insipidus. Based on his moribund prognosis, all

therapeutic interventions were discontinued and the patient soon

died.

Discussion

The occurrence of SAH during infectious endocarditis or as the

presenting symptom of endocarditis is rare. An extended Pubmed

literature search with search terms ‘endocarditis’, ‘subarachnoid

hemorrhage’, ‘infectious intracranial aneurysm’, and ‘mycotic

aneurysm’ revealed 50 cases amongst adults in the English-

language literature since 1950, most of which were case reports.

In only 11 of all 50 cases, SAH was the presenting symptom of

endocarditis. Results are displayed in Table 1.

Although the timeframe in our case report strongly suggests an

association between the occurrence of the SAH and the episode

of endocarditis, pathological proof is lacking. In both cases,

aneurysms were visualized. However, in the fi rst case a specimen

for pathology could not be obtained without jeopardizing the

patient. In the second case the aneurysm was removed and

examined. Pathological examination showed clotted blood

and fi brin without evident signs of infl ammation. However, this

occurred after 26 days of antibiotic treatment. On reviewing the

literature thoroughly, it is evident that the diffi culty in obtaining

pathological proof of these infectious intracranial aneurysms is

omnipresent. Of the 50 cases of SAH, the aneurysms could be

visualized in only 35 cases. Furthermore, in only eight of these

50 cases of SAH, could the diagnosis of infectious intracranial

aneurysms be truly established on the basis of pathological

evidence (see Table 1). In the majority of the studies examined,

the diagnosis of infectious intracranial aneurysms is made on the

basis of the presence of two criteria: the presence of an aneurysm

and endocarditis that has been proven by positive blood cultures

and echocardiography. Our cases do fulfi l these criteria and

therefore we regard these cases as an SAH caused by rupture

of infectious intracranial aneurysms. An infectious intracranial

aneurysm can be caused by micro-embolism to the vasa

vasorum (predominantly streptococcus viridians), or by a septic

embolus wedged in the lumen of a blood vessel (predominantly

staphylococcus aureus). Colonizing micro-organisms might cause

an acute infl ammation of the adventitia with subsequent spread

to a localized necrotizing panarteritis. Weakening, dilatation of

the wall and thus formation of the aneurysm can be the result

[2]. Bacteraemia with seeding of a pre-existent cranial aneurysm

is another possible mechanism. The origin of these emboli and

bacteraemia is most often found in cardiac valve vegetations [3].

Several studies showed a signifi cantly higher number of distally

located aneurysms in the middle cerebral artery in patients with

endocarditis, as in our two cases, as opposed to aneurysms

due to other causes [3-5]. In up to 40% of the cases, SAH is

preceded by a neurological prodrome, most commonly a focal

defi cit caused by embolism [6].

Figure 1a. CT-scan of the brain, transverse section. Intraparen-

chymal and subarachnoid hemorrhage of the right hemisphere

with midline shift and compression of the ventricles.

Figure 1b. Angiography. Distally located on the M3 branch is a

aneurysm visible (arrow).

Figure 2a. CT-scan of the brain, transverse section. Subdural,

intraparenchymal and subarachnoid hemorrhage of the right

hemisphere with midline shift and compression of the ventricles.

Figure 2b. CT-scan of the brain, transverse section. Massive

infarction and edema of the right hemisphere with midline shift

and compression of the ventricles after right-sided hemicraniec-

tomy. Indicated with an arrow is the clip placed on the right a.

cerebri media.

A AB B

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A subarachnoid haemorrhage as a complication of bacterial endocarditis

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Even though SAH heralding endocarditis is a rare phenomenon,

the time frame in the second case is even more unusual. The

second patient was diagnosed with endocarditis and developed

an SAH on the 27th day of antibiotic treatment. SAH due to

infectious aneurysm after effective treatment with antibiotics

has only been described in the literature three times before [7-9].

However, several studies using repeated angiography show that

in about 10% of the cases of infectious intracranial aneurysms

treated with antibiotics, the aneurysm failed to respond to therapy

[9-11]. Not only does the occurrence of neurological symptoms

add to the morbidity of the disease, but also and more importantly,

the mortality of endocarditis has been shown to increase

up to threefold , from 30-50% up to 90% due to neurological

complications [3,12-15,16]. Based on this increased mortality

and morbidity, the recently published guideline for infective

endocarditis strongly advises that additional imaging studies

be performed in patients with endocarditis and neurological

symptoms [16]. There is consensus that a ruptured mycotic

aneurysm should be treated with antibiotics and endovascular

therapy or surgery [17]. However, no such consensus exists on

how to treat an unruptured mycotic aneurysm. On the basis of

a small case series expert opinion advises long term antibiotic

treatment and serial angiography to monitor the effectiveness of

this treatment. Only if the aneurysm is very large or either not

resolving or enlarging despite antibiotics, should the patient be

additionally treated with endovascular therapy or surgery [17,18].

In conclusion we want to state that SAH can be a dangerous

complication of endocarditis that is known to increase morbidity

and mortality. Using these two cases, we want to create awareness

that the formation of infectious intracranial aneurysms and SAH

can occur during an episode of endocarditis. Neurological

symptoms during an episode of endocarditis should always

prompt thorough neurological investigations. Furthermore, since

SAH can be the first presenting symptom of endocarditis, a

diagnostic workup for endocarditis is justified in SAH patients

presenting with fever upon admission.

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