The Open Neuroimaging Journal




ISSN: 1874-4400 ― Volume 13, 2019
CASE REPORT

Convexity Subarachnoid Hemorrhage, Pseudomonas Aeruginosa (PA) Infective Endocarditis and Left Atrial Appendage Occluder (LAAO) Device Infection. A Case Report



Monique Boukobza1, *, Ibtissem Smaali1, Xavier Duval2, Jean-Pierre Laissy3
1 Department of Radiology, Assistance Publique-Hôpitaux de Paris, Bichat Hospital, Paris, France
2 Department of Infectious Diseases, Assistance Publique-Hôpitaux de Paris, Bichat Hospital, INSERM Clinical Investigation Center 007, (S.T., X.D.) and INSERM U738, (C.L., X.D.) Université Paris Diderot, Sorbonne Paris Cité, France
3 Department of Radiology, Assistance Publique-Hôpitaux de Paris, Paris, France; INSERM U1148, Paris, France; University Paris 7, Bichat Hospital, Paris, France

Abstract

An 83 year-old-man with left atrial appendage occluder (LAAO) developed Pseudomonas Aeruginosa (PA) infective endocarditis. MRI at day 3 of onset showed distal small infarcts in both middle cerebral arteries and left postero-inferior cerebellar artery territories. MRI at day 6 revealed two sites of convexity subarachnoid hemorrhage (cSAH). MRA and CTA failed to reveal a Mycotic aneurysm.

The radiologic findings favor the assumption of necrosis of distal branches of mca or of pial arteries wall.

This case present three unusual features: the presence of localized cSAH after initiation of antibiotherapy without mycotic aneurysm being individualized; the late occurrence of infective endocarditis after LAAO implantation; the very rare occurrence of PA in prosthetic infections.

Keywords: Infective Endocarditis, Convexity subarachnoid hemorrhage, T2-GRE images, Mycotic aneurysm, Pseudomonas Aeruginosa, Left atrial appendage occluder.


Article Information


Identifiers and Pagination:

Year: 2017
Volume: 11
First Page: 26
Last Page: 31
Publisher Id: TONIJ-11-26
DOI: 10.2174/1874440001711010026

Article History:

Received Date: 18/01/2017
Revision Received Date: 07/03/2017
Acceptance Date: 20/04/2017
Electronic publication date: 22/05/2017
Collection year: 2017

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© 2017 Boukobza et al.

open-access license: This is an open access article distributed under the terms of the Creative Commons Attribution 4.0 International Public License (CC-BY 4.0), a copy of which is available at: https://creativecommons.org/licenses/by/4.0/legalcode. This license permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.


* Address correspondence to this author at the Department of Radiology, Assistance Publique-Hôpitaux de Paris, Bichat Hospital, 46 rue Henri Huchard, 75018, Paris, France, Tel:+ 33624652932; Fax:+ 33140258305; E-mails: monique.boukobza@aphp.fr; m.boukobza@orange.fr




INTRODUCTION

Subarachnoid hemorrhage (SAH) complicating a mycotic aneurysm (MA) has been reported in only 2- 5% of patients with infective endocarditis (IE) [1Goulenok T, Klein I, Mazighi M, et al. Infective endocarditis with symptomatic cerebral complications: contribution of cerebral magnetic resonance imaging. Cerebrovasc Dis 2013; 35(4): 327-36.
[http://dx.doi.org/10.1159/000348317] [PMID: 23615478]
], while SAH occuring in the absence of an MA remains an infrequent feature of IE [2Krapf H, Skalej M, Voigt K. Subarachnoid hemorrhage due to septic embolic infarction in infective endocarditis. Cerebrovasc Dis 1999; 9(3): 182-4.
[http://dx.doi.org/10.1159/000015952] [PMID: 10207213]
-5Graff-Radford J, Fugate JE, Klaas J, Flemming KD, Brown RD, Rabinstein AA. Distinguishing clinical and radiological features of non-traumatic convexal subarachnoid hemorrhage. Eur J Neurol 2016; 23(5): 839-46.
[http://dx.doi.org/10.1111/ene.12926] [PMID: 26910197]
].

We report a case of an IE of the left atrial appendage occluder (LAAO), caused by Pseudomonas Aeruginosa (PA) and complicated by cerebral ischemia and 2 sites of convexity SAH (cSAH).

CASE REPORT

An 83 year-old-male presented to the emergency room with fever and confusion. He had a stable ischemic heart disease, permanent atrial fibrillation (AF). After an unsuccessful radiofrequency ablation of AF three years earlier, he had an endovascular occlusion of the left atrial appendage (LAA) 30 months before and was left on antiplatelet therapy (lysine acetylsalicylate, 75mg/day).

At admission, confusion, neck stiffness, left sided hemiparesis and endophtalmia with purulent right eye secretions were noted. Body temperature was 40°C and arterial pressure was 153/93. A systolic aortic murmur without abolition of B2 and a systolic mitral murmur were present. An arbitrary antibiotherapy was started on (cefotaxim + ciprofloxacin + amikacin).

The initial work-up revealed in the peripheric blood a WBC count of 9200/mm3, an Hb of 10g/dl, a platelet count of 165 000G/l, a CRP at 182 mg/l, and an isocoagulant status (INR: 1.26, CTA: 1.19). PA was detected in blood cultures (time of positivity after inoculation of 10 and 15 hours).CSF showed a cell count of 40 cells/µl (90% of neutrophils), and rare colonies of PA.

Non-enhanced Brain CT at admission was normal. MRI at day 3 showed multiple small foci of ischemic lesions within the distal middle cerebral arteries (MCA) territories, the pons and the left postero-inferior cerebellar artery territory Fig. (1-a-b). There was no hemorrhagic lesion.

Transthoracic echography (TTE) and transesophageal echography (TEE) showed a huge mobile tissular lesion located on the LAAO, corresponding either to septic vegetation or to thrombi. The mitral leaflets were heavily calcified

A definite IE by PA – according to the modified Duke’s criteria - complicated with endophtalmia, cerebral emboli and meningitis was diagnosed and adequate antibiotherapy was started on.

Repeat MRI at day 6 showed besides the lesions described before, two sites of localized SAH on FLAIR and T2-GRE images at the left temporal convexity -extending to the external part of the sylvian fissure- and at a right temporal sulcus, and revealed a minor hemorrhagic transformation of the rolandic infarct Figs. (1-c-d-e) 3D-TOF MR Angiography of the Willis circle showed a dolicho-basilar trunk.

Brain CT-Angiogram (CTA) failed to reveal an MA or other cause of SAH, and showed a stenosis (50%) of both internal carotid arteries.

The 18-F-FDG PET scan was compatible with the diagnosis of IE of the LAAO with septic localizations at the right eye.

Per-operative TEE confirmed the diagnosis of a huge plurilobular formation fixed to the LLAO, a moderate to severe mitral regurgitation because of a P2-A2 mitral prolapse and images of small vegetations on P2. The LAAO and the native mitral valve apparatus were removed and a mitral biological prosthesis was implanted.

The patient was rapidly discharged from the operative room with stable hemodynamics. However, he died 3 days later from a refractory cardiogenic shock.

Fig. (1)
Axial DWI shows acute small embolic infarcts in territories of both middle cerebral arteries distal territory (a) and of the left postero-inferior cerebellar artery (a-b). Axial FLAIR, fluid attenuated inversion recovery (c) and axial T2 GRE images (d-e) show acute SAH filling partially the left sylvian fissure, the adjacent temporal convexity and a small sulcus at the right temporal convexity.


DISCUSSION

The reported case presents three unusual features: firstly, the presence of localized cSAH without MA being individualized; secondly the late occurrence of IE after LAAO implantation; finally PA being an infrequent bacterium responsible for prosthetic infections.

SAH without associated MA has been rarely reported in IE.

Krapf [2Krapf H, Skalej M, Voigt K. Subarachnoid hemorrhage due to septic embolic infarction in infective endocarditis. Cerebrovasc Dis 1999; 9(3): 182-4.
[http://dx.doi.org/10.1159/000015952] [PMID: 10207213]
] reported a case of IE with an abundant amount of blood in the sylvian fissure, and thirteen days later, the patient developed a huge parenchymal hematoma. At surgery, the proximal segment of the MCA was necrotic, and no MA was identified. A follow-up angiography did not reveal any vascular lesion. The patient survived with a left sided deficit.

Chukwudelunzua [3Chukwudelunzu FE, Brown RD Jr, Wijdicks EF, Steckelberg JM. Subarachnoid haemorrhage associated with infectious endocarditis: case report and literature review. Eur J Neurol 2002; 9(4): 423-7.
[http://dx.doi.org/10.1046/j.1468-1331.2002.00432.x] [PMID: 12099929]
] identified 8 cases of SAH among 489 patients with IE (1%), diagnosed by Brain-CT. Cerebral angiography or autopsy did not reveal any MA in 6 cases. SAH was localized at the fronto-parietal sulci in 3, at the sylvian fissure in 2 and at the basal cisterns in1. In 1 /29 cases of spontaneous localized cSAH the SAH was related to IE, furthermore associated with cerebral microbleeds [6Hochberg FH, Fisher CM, Roberson GH. Subarachnoid hemorrhage caused by rupture of a small superficial artery. Neurology 1974; 24: 319-21.]. Recently, Graff-Radford [5Graff-Radford J, Fugate JE, Klaas J, Flemming KD, Brown RD, Rabinstein AA. Distinguishing clinical and radiological features of non-traumatic convexal subarachnoid hemorrhage. Eur J Neurol 2016; 23(5): 839-46.
[http://dx.doi.org/10.1111/ene.12926] [PMID: 26910197]
] reported a prevalence of 10% of IE in a series of 88 patients presenting with non-traumatic cSAH.

SAH are usually attributed to ruptured MA, even when no aneurysm is demonstrable because they are sometimes obliterated by the hemorrhage that they produce, hence their angiographic and even pathologic demonstration is not always possible.

Our patient had no image confirming rupture of MA on CTA. The SAH was mild to hide any aneurysm and the patient was only under antiplatelet therapy, ruling out any suspicion of anticoagulation overdose responsible for the bleeding.

Septic emboli can be responsible for erosive arteritis causing rupture of the artery wall and intracerebral hemorrhage. This eventuality occurs during uncontrolled infection, particularly with virulent organisms such as PA, which can be the case of our patient who continued to have septic signs although antibiotherapy had been instituted.

Furthermore, in the current case, the concomitance of multiple distal infarcts and 2 sites of cSAH lead us to believe that an erosive arteritis secondary to the bacterial infection was responsible for wall rupture of distal arteries. Nevertheless, rupture of small pial artery as a cause of SAH had been previously demonstrated in an autopsy report [7Kumar S, Goddeau RP Jr, Selim MH, et al. Atraumatic convexal subarachnoid hemorrhage: clinical presentation, imaging patterns, and etiologies. Neurology 2010; 74(11): 893-9.
[http://dx.doi.org/10.1212/WNL.0b013e3181d55efa] [PMID: 20231664]
]. Rupture of small pial arteries might also be responsible of cSAH in the setting of IE.

Furthermore, CAA is the most commonly reported cause of cSAH and in this eventuality, the most common location for cSAH is the fronto-parietal area [8Wilson D, Hostettler IC, Ambler G, Banerjee G, Jäger HR, Werring DJ. Convexity subarachnoid haemorrhage has a high risk of intracerebral haemorrhage in suspected cerebral amyloid angiopathy. J Neurol 2017; 264(4): 664-73.
[http://dx.doi.org/10.1007/s00415-017-8398-y] [PMID: 28154972]
]. In reversible vasoconstriction syndrome (RVCS), the second commonest cause as in cerebral venous thrombosis (CVT) and PRES, cSAH may be extensive along both convexities [9Ducros A, Fiedler U, Porcher R, Boukobza M, Stapf C, Bousser MG. Hemorrhagic manifestations of reversible cerebral vasoconstriction syndrome: frequency, features, and risk factors. Stroke 2010; 41(11): 2505-11.
[http://dx.doi.org/10.1161/STROKEAHA.109.572313] [PMID: 20884871]
-11Hefzy HM, Bartynski WS, Boardman JF, Lacomis D. Hemorrhage in posterior reversible encephalopathy syndrome: imaging and clinical features. AJNR Am J Neuroradiol 2009; 30(7): 1371-9.
[http://dx.doi.org/10.3174/ajnr.A1588] [PMID: 19386731]
]. In the reported case, cSAH was present at both temporal convexities. Further studies are necessary to evaluate the occurrence and the imaging features of cSAH related to IE.

On the other hand, this patient was followed for ischemic heart disease and was under aspirin for long years. He had never presented hypersensitive clinical manifestations and no worsening of his coronary artery disease. That why laboratory tests were not indicated for him. Recent studies have shown that only short-term use of low-dose aspirine is associated with increased risk of SAH [12Phan K, Moore JM, Griessenaue CJ, et al. Aspirine and risk of subarachnoid hemorrhage. Stroke 2017; 48 [Epub ahead of print]].

IE located at the LAAO is an extremely rare complication. Two recent studies about feasibility and safety with the fourth generation watchman LAAOs failed to detect device infection [13López Mínguez JR, Asensio JM, Gragera JE, et al. Two-year clinical outcome from the Iberian registry patients after left atrial appendage closure. Heart 2015; 101(11): 877-83.
[http://dx.doi.org/10.1136/heartjnl-2014-306332] [PMID: 25573984]
]. Only one case of infection associated with atrial appendage occluder, occurring a few days after implantation has been reported (staphylococcus aureus) and was related to lack of sterile conditions during the procedure [9Ducros A, Fiedler U, Porcher R, Boukobza M, Stapf C, Bousser MG. Hemorrhagic manifestations of reversible cerebral vasoconstriction syndrome: frequency, features, and risk factors. Stroke 2010; 41(11): 2505-11.
[http://dx.doi.org/10.1161/STROKEAHA.109.572313] [PMID: 20884871]
]. IE due to gram-negative bacilli (GNB) represents 4% of all IE and PA accounts for only about 3% of IE due to GNB [14Khumri TM, Thibodeau JB, Main ML. Transesophageal echocardiographic diagnosis of left atrial appendage occluder device infection. Eur J Echocardiogr 2008; 9(4): 565-6.
[PMID: 17681493]
].

PA endocarditis is usually associated with the use of intravenous drugs and with prosthetic devices [15Loubet P, Lescure FX, Lepage L, et al. Endocarditis due to gram-negative bacilli at a French teaching hospital over a 6-year period: clinical characteristics and outcome. Infect Dis (Lond) 2015; 47(12): 889-95.
[http://dx.doi.org/10.3109/23744235.2015.1075660] [PMID: 26260729]
]. PA infects mainly elderly immunodepressive patients and is frequent in intensive care unit. We suspected that our patient contracted the PA either from a recent stay at hospital after blood transfusion, or via ocular contamination, as he had a severe endophtalmia.

To the best of our knowledge, this is the first reported case of localized SAH secondary to PA IE in a patient after LAAO system implantation.

CONCLUSION

Localized cSAH without MA and in the absence of anticoagulation is a very rare neurological complication of IE.

This case of late prosthetic IE demonstrates that patients with LAAO are at risk to develop infection of their device and should benefit from the same preventive recommendations as the high –risk populations.

Finally, localized cSAH should suggest IE when occurring in patients with implanted heart devices.

CONFLICT OF INTEREST

The authors confirm that this article content has no conflict of interest.

ACKNOWLEDGEMENTS

Declared none.

REFERENCES

[1] Goulenok T, Klein I, Mazighi M, et al. Infective endocarditis with symptomatic cerebral complications: contribution of cerebral magnetic resonance imaging. Cerebrovasc Dis 2013; 35(4): 327-36.
[http://dx.doi.org/10.1159/000348317] [PMID: 23615478]
[2] Krapf H, Skalej M, Voigt K. Subarachnoid hemorrhage due to septic embolic infarction in infective endocarditis. Cerebrovasc Dis 1999; 9(3): 182-4.
[http://dx.doi.org/10.1159/000015952] [PMID: 10207213]
[3] Chukwudelunzu FE, Brown RD Jr, Wijdicks EF, Steckelberg JM. Subarachnoid haemorrhage associated with infectious endocarditis: case report and literature review. Eur J Neurol 2002; 9(4): 423-7.
[http://dx.doi.org/10.1046/j.1468-1331.2002.00432.x] [PMID: 12099929]
[4] Mangla R, Drumsta D, Alamst J, Mangla M, Potchen M. Cerebral convexity subarachnoid hemorrhage: various causes and role of diagnostic imaging. Emerg Radiol 2015; 22(2): 181-95.
[http://dx.doi.org/10.1007/s10140-014-1251-z] [PMID: 25001597]
[5] Graff-Radford J, Fugate JE, Klaas J, Flemming KD, Brown RD, Rabinstein AA. Distinguishing clinical and radiological features of non-traumatic convexal subarachnoid hemorrhage. Eur J Neurol 2016; 23(5): 839-46.
[http://dx.doi.org/10.1111/ene.12926] [PMID: 26910197]
[6] Hochberg FH, Fisher CM, Roberson GH. Subarachnoid hemorrhage caused by rupture of a small superficial artery. Neurology 1974; 24: 319-21.
[7] Kumar S, Goddeau RP Jr, Selim MH, et al. Atraumatic convexal subarachnoid hemorrhage: clinical presentation, imaging patterns, and etiologies. Neurology 2010; 74(11): 893-9.
[http://dx.doi.org/10.1212/WNL.0b013e3181d55efa] [PMID: 20231664]
[8] Wilson D, Hostettler IC, Ambler G, Banerjee G, Jäger HR, Werring DJ. Convexity subarachnoid haemorrhage has a high risk of intracerebral haemorrhage in suspected cerebral amyloid angiopathy. J Neurol 2017; 264(4): 664-73.
[http://dx.doi.org/10.1007/s00415-017-8398-y] [PMID: 28154972]
[9] Ducros A, Fiedler U, Porcher R, Boukobza M, Stapf C, Bousser MG. Hemorrhagic manifestations of reversible cerebral vasoconstriction syndrome: frequency, features, and risk factors. Stroke 2010; 41(11): 2505-11.
[http://dx.doi.org/10.1161/STROKEAHA.109.572313] [PMID: 20884871]
[10] Boukobza M, Crassard I, Bousser MG, Chabriat H. Radiological findings in cerebral venous thrombosis presenting as subarachnoid hemorrhage: a series of 22 cases. Neuroradiology 2016; 58(1): 11-6.
[http://dx.doi.org/10.1007/s00234-015-1594-5] [PMID: 26376804]
[11] Hefzy HM, Bartynski WS, Boardman JF, Lacomis D. Hemorrhage in posterior reversible encephalopathy syndrome: imaging and clinical features. AJNR Am J Neuroradiol 2009; 30(7): 1371-9.
[http://dx.doi.org/10.3174/ajnr.A1588] [PMID: 19386731]
[12] Phan K, Moore JM, Griessenaue CJ, et al. Aspirine and risk of subarachnoid hemorrhage. Stroke 2017; 48 [Epub ahead of print]
[13] López Mínguez JR, Asensio JM, Gragera JE, et al. Two-year clinical outcome from the Iberian registry patients after left atrial appendage closure. Heart 2015; 101(11): 877-83.
[http://dx.doi.org/10.1136/heartjnl-2014-306332] [PMID: 25573984]
[14] Khumri TM, Thibodeau JB, Main ML. Transesophageal echocardiographic diagnosis of left atrial appendage occluder device infection. Eur J Echocardiogr 2008; 9(4): 565-6.
[PMID: 17681493]
[15] Loubet P, Lescure FX, Lepage L, et al. Endocarditis due to gram-negative bacilli at a French teaching hospital over a 6-year period: clinical characteristics and outcome. Infect Dis (Lond) 2015; 47(12): 889-95.
[http://dx.doi.org/10.3109/23744235.2015.1075660] [PMID: 26260729]

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