The first described case of late infective endocarditis after implantation of Figulla Flex Ⅱ ASD occluder

Abstract

Infective endocarditis is one of the complications following the percutaneous occlusion of an atrial septal defect (ASD) with a closure device. To the best of our knowledge, no case reports have been published of infective endocarditis associated with the Figulla Flex Ⅱ ASD occluder (FSO; Occlutech GmbH, Jena, Germany). We present the case of a 50-year-old woman who underwent a transcatheter closure of an ASD with FSO almost 2 years prior to presentation to our institution. Echocardiography showed a mobile vegetation (20 × 10 mm), and her blood culture grew β-hemolytic streptococci. Magnetic resonance imaging revealed acute cerebral infarction. Those findings were diagnosed as late infective endocarditis associated with the ASD closure device. The patient was treated with antibiotics and underwent surgical removal of the FSO, which showed incomplete endothelialization, and surgical repair of ASD. After surgery, the patient made a complete recovery without complications or residual shunts. This case highlights the risk of late infective endocarditis in patients after closure of ASD with an FSO with incomplete endothelialization.

<Learning objective: Endothelialization of Figulla Flex Ⅱ ASD occluder (FSO) devices is presumed to be complete within 3 to 6 months. However, some patients have presented with poor endothelialization of the device. In patients who undergo percutaneous atrial septal defect closure with an FSO, delayed endothelialization of the device could be a risk for late infective endocarditis. We suggest the need to be aware of the onset of late infective endocarditis even after 6 months after placement of an FSO.>

Introduction

A secundum atrial septal defect (ASD Ⅱ) is a common congenital disease, and transcatheter closure has become the standard approach for the disease. Today, the most frequently used devices in Japan are the Figulla Flex Ⅱ ASD occluder (FSO; Occlutech GmbH, Jena, Germany) and the Amplatzer septal occluder (ASO; Abbott, St. Paul, Minnesota, USA). The FSO device is constructed from 0.082 to 0.186 mm nitinol wires tightly woven into 2 discs with a connecting waist. The prosthesis is filled with a polyester patch (30–45 µm thick). Previous studies have shown that the differences between the complications and outcomes after placement of either the ASO or FSO devices were not significant [1, 2]. The complications following closure of an ASD with an FSO or ASO include device embolization, cardiac perforation/erosion/rupture, cardiac tamponade, endocarditis, thromboembolic events, cardiac arrhythmias, headache, migraine, and hematoma. Late bacterial endocarditis has been the focus of some case reports of patients who underwent implantation of an ASO to repair an ASD [3, 4]. However, not much information is available regarding late infective endocarditis associated with an FSO.

Case report

A 50-year-old woman was urgently transferred to our hospital because of an acute cerebral infarction without neurological signs/symptoms and a splenic infarction after a 12-day history of intermittent fevers up to 38 °C, arthralgias, headache, an Osler node (single, small, tender node) (Fig. 1) on her right palm, and Janeway lesions on her palms and soles (multiple, small, nontender lesions). She had a history of ASD Ⅱ that had been repaired percutaneously with a 30-mm FSO 21 months prior to her admission. Transesophageal echocardiography (TEE) had revealed an ASD Ⅱ in anterosuperior area of atrial septum, which measured 27.3 × 11.6 mm without rim deficiency. The previous device closure was percutaneously performed under intracardiac echocardiographic guidance. The defect of ASD Ⅱ was sized with a 34-mm sizing balloon. A 30-mm FSO was selected based on the measurement of the stop-flow method, which measured 26.4 mm. The procedure was successful without any residual shunts, which were confirmed by TEE. In addition, transthoracic echocardiography did not reveal the presence of residual shunts 1 month, 3 months, and 12 months after the repair. She had no past medical history including immuno-deficiency and metallic allergy other than ASD.

Fig. 1.

Osler's node (arrow) on the right fourth finger.

On admission, laboratory testing revealed elevated C-reactive protein (14.9 mg/dL), procalcitonin (1.23 ng/ml), white blood cell (9000 /µl), and D-dimer (2.3 µg/mL) levels. Transthoracic and transesophageal echocardiography showed a 20 × 10-mm mobile vegetation attached to the left side of the prosthetic device located in the inter-atrial septum (Fig. 2, Online Video S1, S2), which did not compromise the tricuspid and mitral valves. She received empiric antimicrobial therapy (vancomycin and gentamicin) based on the current guidelines. Although her blood cultures were positive for β-hemolytic streptococci, she denied a history of wounds on her body or dental procedures that involved gingival tissue or oral mucosa.

Fig. 2.

Transthoracic (A) and transesophageal (B) echocardiograms reveal vegetation (arrows).

LA, left atrium; RA, right atrium; LV, left ventricular.

The FSO was thought to be infected and a possible source of multiple systemic emboli and persistent bacteremia. We decided on removing the device surgically as soon as possible because of the large vegetation with a risk of systemic emboli. The infected FSO was removed on day 3 of hospitalization through a right atrial approach. Both the right and left discs of the device showed poor endothelialization (Fig. 3). There were also vegetations on both the right and left sides of the removed FSO device (Fig. 3). Other cardiac structures including the valves appeared normal. The residual ASD Ⅱ was repaired by a patch. The culture of vegetations on the device was negative probably because of antibiotic therapy.

Fig. 3.

Intraoperative findings (A, right-atrial-side of the device) and removed device (B, right atrial side; C, left atrial side of the device) include incomplete endothelialization on the surface of device and vegetation (arrow).

The patient was treated with 6 weeks of ampicillin and 2 weeks of gentamicin, postoperatively. She was discharged home with no complications or residual shunts on the 45th hospital day.

Discussion

Here, we report a case of late infective endocarditis associated with an FSO device. Transcatheter closure has recently become the preferred intervention for repair of ASD Ⅱ. Cases of endocarditis associated with device closure of ASDs have not been reported by large studies that included a total of 1315 patients with mean follow-ups ranging from 1 to 3.6 years [1, 2, 5]. To our knowledge, this is the first case report of infective endocarditis involving an FSO device, although some cases of endocarditis involving other closure devices for ASD have been reported [3, 4].

Because endocarditis associated with ASDs are rare, prophylaxis for infective endocarditis is not generally recommended even after dental procedures. However, some guidelines suggest that antibiotic prophylaxis for 6 months after implantation is recommended for patients undergoing percutaneous ASD closure. The evidence for this recommendation is not sufficient, and investigation of an animal model found that the early device that was used in the study underwent complete endothelialization by 3 months after implantation [6]. Therefore, the appropriate duration of prophylaxis for bacterial endocarditis in patients after receiving an FSO device remains unclear. Additionally, the formation of vegetations reportedly involve not only bacteremia but also the formation of thrombus. Although antiplatelet drug administration for 6 months is generally used, the prophylactic use of antiplatelet drug for a period exceeding 6 months may be expected to prevent infective endocarditis in patients with incomplete endothelialization. Some authors have also proposed that the incomplete endothelialization of a device used to repair an ASD is involved in the development of late infective endocarditis because the exposure of metal coils due to incomplete endothelialization promotes the formation of a thrombus and infective endocarditis [3, 4]. Actually, although the FSO device had been implanted almost 2 years before the patient's onset of endocarditis, endothelialization of the device was poor. However, it is difficult to estimate what is the risk factor of incomplete endothelialization. According to a past report about late infective endocarditis related to percutaneous ASD closure, implanted closure devices are relatively large in patients with incomplete endothelialization after elapsing more than 6 months [4]. Therefore, we may need to pay attention to patients with relatively large closure devices. On the other hand, the manufacturer proposes that FSO device decreases the nickel toxicity and thrombogenicity and promotes neoendothelialization due to absence of hub, coting with titanium, and a lower metal load [7]. Because patients with delayed endothelialization of devices could be at risk for late infective endocarditis after FSO, we need an imaging technique that could estimate the degree of neoendothelialization of the device. Also, until additional evidence is available on the duration of prophylaxis, we might consider prescribing a prolonged course of antibiotics and antiplatelet drug to prevent infective endocarditis after FSO implantation.

The cause of infective endocarditis in early phase after device implantation may be bacterial inoculation during the procedure. On the other hand, late infective endocarditis associated with device is often secondary to hematogenously disseminated infection and caused by transient bacteremia during daily life. In the present case, blood cultures were positive for β-hemolytic streptococci, which cause a wide variety of infections including cellulitis, necrotizing fasciitis, bacteremia, and infective endocarditis [8]. However, our patient did not have a history of wounds on her body, dental procedures, or other infections. Moreover, although infective endocarditis due to β-hemolytic streptococci is relatively uncommon, it can be life-threatening because of high rate of cardiac valve destruction, cardiac abscess formation, and systemic embolization [8].

In this case, we decided to perform early surgery despite the presence of acute cerebral infarction. Cerebral infarction is a common complication and is associated with increased mortality in patients with infective endocarditis [9]. However, the optimal time to perform surgery on patients with infected endocarditis with cerebral infarction remains controversial. Although the relationship between cerebral bleeding and cardiopulmonary bypass on acute cerebral infarction is still not clear, some reports have demonstrated early surgery improved clinical results without increasing the events of neurological complications [9]. In addition, Athan et al. reported that early removal of the device was associated with improved survival at 1 year [10].

We reported a rare case of endocarditis involving an FSO device with incomplete endothelialization. We suggest that physicians should be aware that late infective endocarditis of an FSO device can occur later than 6 months after placement of an FSO device to repair an ASD.

Declaration of Competing Interest

The authors declare that there are no conflicts of interest regarding the publication of this article.