Copyright © 2006, The European Society of Cardiology
Inflammatory, abscess-forming foreign body reaction mimics a thrombus formation on an atrial septal defect closure device: A commented case report
aHannover Medical School, Department of Cardiology and Angiology, Carl-Neuberg-Strasse 1, 30625 Hannover, Germany
bDepartment of Pathology, Hannover Medical School, Carl-Neuberg-Strasse 1, 30625 Hannover, Germany
Received 27 January 2006; received in revised form 21 March 2006; accepted after revision 30 March 2006.
* Corresponding author. Tel.: +49 511 532 2532; fax: +49 511 532 3357. divchev.dimitar{at}mh-hannover.de
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Abstract |
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We are presenting a case of floating left and right atrial formations on an atrial septal defect occluder system (23mm StarFLEX®-Occluder) initially supposed to be thrombotic appositions in a 57-year-old man. The closure was performed on the background of left hemispheric stroke and atrial septal aneurysm (ASA) with patent foramen ovale (PFO). The suspect structures were detected in the 6-month follow-up by transesophageal echocardiography (TEE). The patient underwent a successful surgical explantation of the closure device and closure of the patent foramen ovale (PFO) using a pericardial patch. The pathological evaluation of the biatrial device associated appositions revealed hytrophic heart muscle tissue with perifocal scarring and purulent abscess-forming, granulating and foam-cell including inflammatory foreign body reaction instead of the expected thrombus formation.
Keywords: TEE, transesophageal echocardiography; ASD, atrial septal defect; PFO, patent foramen ovale; ASA, atrial septal aneurysm; INR, international normalized ratio
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Case presentation |
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A 57-year-old man was admitted for transcatheter closure of a patent foramen ovale (PFO).
He had a medical history of left hemispheric stroke 1 month prior to presentation for further evaluation in our department. Hypercoagulation as well as other extracardial causes of systemic embolism such as significant carotid plaques were excluded. There were no mentionable cardiovascular risk factors as well as no signs of arrhythmia. Conventional electrocardiography and Holter's ECG showed sinus rhythm. The patient did never have palpitations, angina or symptoms of heart failure. Transesophageal echocardiography revealed a hypermobile atrial septum fulfilling the criteria of an atrial septal aneurysm (bidirectional excursion of 20mm) with minimal separation at typical position for a PFO. Right heart contrast agent injection demonstrated a significant right to left shunt under Valsalva's maneuver.
The patient underwent a TEE-guided transcatheter closure of the patent foramen ovale in local anaesthesia using a 23mm StarFLEX®-Occluder without residual shunt. He received anticoagulation with intravenous heparin during and in the first days after PFO closure. Phenprocoumon was started and continued for 6 months with a target INR of 2.5 together with aspirin 100mg per day in order to avoid device associated thrombus formation. He received 2g Cefazolin intravenously during the procedure.
A scheduled follow-up was planed after 6 months.
The patient returned to an early follow-up after 2 months. He reported paroxysmal episodes of palpitations. The general physician registered new onset intermittent atrial fibrillation in several ECG. Additional TEE did not show any complications after closure device implantation to follow-up (Fig. 1). There was no residual shunt in colour Doppler imaging as well as after right heart contrast agent injection. In contrast, regular TEE after 6 months showed floating echogenic structures connected to the left and right atrial parts of the StarFLEX®-Occluder without any additional atrial or ventricular thrombi and absence of spontaneous echo contrast (Figs. 2 and 3
). There were no signs of systemic embolism. PW-Doppler evaluation showed normal flow velocities (60cm/s) within the left atrial auricle. The patient did not report any more palpitations after early follow and ECG showed sinus rhythm. There were no signs of systemic inflammation after device implantation to follow-up. INR constantly ranged between 2.0 and 3.0.
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The patient was referred to surgery and underwent a successful explantation of the device together with the appositions and a closure of the atrial septal defect using a pericardial patch.
The pathological findings of the biatrial device associated appositions revealed hytrophic heart muscle tissue with perifocal scarring and purulent abscess-forming, granulating and foam-cell including inflammatory foreign body reaction instead of the expected thrombus formation (Figs. 4–7).
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Comment |
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Device associated atrial thrombi are reported complications after transcatheter ASD or PFO closure1–5 and present their selves as solitary or floating appositions at the right and/or left atrial parts of the closure device on TTE and/or TEE. The usual strategy for managing such complications is to resolve the thrombus formation by an accurate further anticoagulation (in cases with only platelet antiaggregation) or even intravenous thrombolysis. Other cases of floating device associated appositions on TEE are immediately referred to surgery concerning systemic embolism or at least after unsuccessful trial to resolve the TEE-detected "thrombus".
Synthetic polymer implants can lead to inflammatory response by interactions of the tissue with the implant. The inflammatory response resembles the well known foreign body reaction. In this sense cellular and molecular components of the specific and nonspecific immune responses are involved. Upon implantation, fibrinogen, among other proteins, adsorbs to the material and provides a substrate for integrin mediated macrophage binding.6–12 It has been shown that type and extent of this immune response strongly depend on the material and its surface characteristics.13–19
The StarFlex Double umbrella is a device with 4- or 6-arm metallic framework, Dacron® fabric and self-centering mechanism achieved by nitinol microsprings.
Dacron®, for instance, has been reported to trigger chronic inflammation in vascular prosthetic materials characterised by the presence of activated macrophages and foreign body giant cells in animal models.6 Proliferation of surrounding fibroblasts accompanies the chronic foreign body response and can eventually lead to excessive encapsulation, causing such devices to fail.6–12
In our case we do not have any strong evidence to suppose the primary occurrence of a biatrial thrombus formation by new onset intermittent atrial fibrillation and secondary superinfection. This is especially uncertain on the background of strictly managed anticoagulation and platelet antiaggregation, absence of systemic inflammation after transcatheter closure and periprocedural application of Cefazolin as well as inconspicuous early TEE follow-up.
Nevertheless, there is no doubt that an infective mechanism must be the reason for a purulent abscess-forming process because a foreign body reaction alone is not associated with appearance of granulocytes and microabscesses. The onset of inflammation remains unclear. From our point of view intravenous antibiotics should be given during implantation although application could not avoid the inflammation in this case. It should be mentioned that there are no data demonstrating the further outcome with or without periprocedural intravenous application of antibiotics.
The further decision for therapy is fundamentally based on TEE, though without any hard echocardiographic criteria to discriminate the real kind and origin of the suspect structures. This case impressively demonstrates the necessity of accurate pathological evaluation of explanted devices in order to estimate an approximate incidence of primary non-thrombotic appositions after closure of atrial septal defects.
It seems rather impossible to predict the appropriate therapy for thrombus like device associated appositions even taking into consideration the whole summary of medical history, laboratory results and imaging methods. Surgery might be offensively discussed in cases with such complications occurring under accurate anticoagulation.
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