A rare case of patent foramen ovale occluder embolization: A sneaky clinical presentation

Luca Allievi; Amedeo Bongarzoni; Guido Tassinario; Barbara Conconi; Gabriele Tumminello; Stefano Carugo

doi:10.4103/ACCJ.ACCJ_11_21

CASE REPORT

Year : 2021 | Volume : 3 | Issue : 2 | Page : 81-84

A rare case of patent foramen ovale occluder embolization: A sneaky clinical presentation

Luca Allievi¹, Amedeo Bongarzoni², Guido Tassinario², Barbara Conconi², Gabriele Tumminello², Stefano Carugo³
¹ Department of Clinical Sciences and Community Health, University of Milan, Milan, Italy
² Department of Cardiology, ASST Santi Paolo e Carlo, University of Milan, Milan, Italy
³ Department of Cardiology, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy

Date of Submission	01-Jun-2021
Date of Decision	07-Jul-2021
Date of Acceptance	08-Jul-2021
Date of Web Publication	08-Dec-2021

Correspondence Address:
Dr. Luca Allievi
Department of Clinical Sciences and Community Health, University of Milan, Via Festa del Perdono 7, 20122 Milan
Italy

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ACCJ.ACCJ_11_21

Abstract

Patent foramen ovale (PFO) is a common congenital defect in adults; its closure with an occluder device is usually a safe procedure. However, a rare and potentially fatal complication is the device embolization. We report a case of a 52-year-old man with bicuspid aortic valve and surgically corrected aortic coarctation, in which a PFO occluder device migrated to the abdominal aorta. We recommend paying attention to the risk factors before the intervention (PFO characteristics and multiple congenital defects) and to the clinical-echocardiographic follow-up, to prevent, recognize, and treat this severe complication as soon as possible.

Keywords: Atrial septal aneurysm, case report, dyspnea, limb pain, patent foramen ovale, transcatheter closure device

How to cite this article:
Allievi L, Bongarzoni A, Tassinario G, Conconi B, Tumminello G, Carugo S. A rare case of patent foramen ovale occluder embolization: A sneaky clinical presentation. Ann Clin Cardiol 2021;3:81-4

How to cite this URL:
Allievi L, Bongarzoni A, Tassinario G, Conconi B, Tumminello G, Carugo S. A rare case of patent foramen ovale occluder embolization: A sneaky clinical presentation. Ann Clin Cardiol [serial online] 2021 [cited 2023 Jun 4];3:81-4. Available from: http://www.onlineacc.org/text.asp?2021/3/2/81/336215

Introduction

Patent foramen ovale (PFO) is a congenital intracardiac defect present in about 25% of the general population, due to incomplete fusion of septum primum and secundum after birth. It predisposes to paradoxical embolism, in which emboli from the peripheral venous system provoke systemic strokes or infarcts. Given the low incidence of paradoxical embolism (1% per year),^[1] percutaneous PFO closure is performed only in the presence of a thromboembolic event (e.g., stroke) caused with high probability by the presence of a PFO associated with a high risk of recurrence.^[2] After the procedure, several complications can occur, as hemorrhage, pulmonary embolism, infective endocarditis, cardiac perforation and device erosion, fracture, thrombosis, or embolization.^[3] Device embolization is very rare (0.1%–0.4%) but potentially life-threatening and requires immediate removal of the embolized device.^[3] Possible migration sites are pulmonary artery, heart chambers, aortic arch, descending thoracic, and abdominal aorta.^[4] We report below a case of embolization of a PFO occluder device into the abdominal aorta.

Case Report

A 52-year-old male patient came to our echocardiography laboratory at the San Carlo Hospital in Milan on December 31, 2020, following the onset of dyspnea with light exertion and chest pain radiated to both upper limbs. He denied having cardiovascular risk factors but reported being a carrier of a bicuspid aortic valve (BAV) and having been operated for aortic coarctation (AC) at the age of 14. In June 2020, he had been admitted to another hospital neurology ward for an ischemic stroke of the vertebrobasilar circulation; the transthoracic echocardiogram (TTE) had shown a slight spontaneous left–right shunt and positive bubble study, with the final diagnosis of PFO, and ascending aorta dilatation. In October 2020, percutaneous PFO closure by endovascular positioning of an Occlutech device (23/25 mm) had been performed; then, he had been discharged on dual antiplatelet and statin therapy without performing a TTE before discharge, only suggesting to perform a TTE with bubble study 4–6 months after the intervention. About 7 days after discharge, upon resuming work (employee in septic service), he started feeling shortness of breath with light efforts and chest pain radiating to the upper limbs (described as myalgia) every day but only upon awakening. For this reason, he tried to contact the hospital where the device was placed, but after 2 months without having been visited, he decided to come to our laboratory for a physical examination with TTE. The physical examination was normal, while the only electrocardiographic finding was a right bundle branch block. The TTE showed the absence of the occluder device in place, with a spontaneous left–right shunt [Figure 1]; in addition, the transesophageal echocardiogram (TOE) detected the absence of the device and the presence of an atrial septal aneurysm (ASA) and a longer tunnel (24 mm) [Figure 2] than it was detected in the previous hospitalization (13 mm). A chest/abdomen computed tomography scan with contrast showed the device into the abdominal aorta, at the first lumbar vertebra (L1), near the origin of the celiac trunk (from which the superior mesenteric artery also arose) and just proximal to the origin of the renal arteries [Figure 3]. An abdominal aorta ultrasound showed the device completely intact (two hemidiscus), blocked into the aortic lumen at the origin of the celiac trunk [Figure 4]: The device partially occluded both vessels without causing gastrointestinal or lower limb symptoms. The patient was then admitted to our cardiology ward, still presenting myalgias during the night and in the morning (no fever, negative severe acute respiratory syndrome coronavirus-2 RT-PCR swab test, and normal creatine kinase). Then, the patient was transferred to the hospital where the device had been deployed and the retrieval was performed on January 5, 2021. Through right femoral access, the device was captured and pulled with a 35 mm Gooseneck snare [Figure 5], losing the device into the right external iliac artery; then, it was captured with a 50 cm bioptome, but the device was lost for a second time into the right femoral artery, from where it was finally decided to surgically remove it. After the device removal, the patient felt no more myalgias. A new PFO closure with NobleStitch was proposed to the patient, but he decided not to undergo another procedure for at least 1–2 months. The patient was discharged on January 8, 2021.

Figure 1: TTE performed from a 2D subcostal view demonstrating the absence of the PFO occluder device in the interatrial septum (Panel A) and the presence of a spontaneous left-to-right shunt due to PFO in CFM (Panel B). 2D: Two-dimensional, CFM: Color-flow mode, LA: Left atrium, LV: Left ventricle, PFO: Patent foramen ovale, RA: Right atrium, RV: Right ventricle, TTE: Transthoracic echocardiogram

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Figure 2: TOE subsequently performed confirming the absence of the occluder device in the interatrial septum and showing a long PFO tunnel (24 mm) in a modified-bicaval view (off-axis view). Ao: Aorta, LA: Left atrium, PFO: Patent foramen ovale, RA: Right atrium, PFO: Patent foramen ovale, TOE: Transesophageal echocardiogram

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Figure 3: Thoracoabdominal computed tomography scan from an axial (Panel A), sagittal (Panel B), and coronal plane (Panel C) showing the occluder device into the abdominal aorta at L1 vertebral level near the origin of the coeliac trunk. Ao: Aorta, L1: First lumbar vertebra

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Figure 4: Abdominal ultrasound demonstrating the device with its two hemidiscus into the aortic lumen (Panel A), blocked at the origin of the celiac trunk as seen in CFM (Panel B). Ao: Aorta, CFM: Color-flow mode

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Figure 5: Fluoroscopic image showing the percutaneous retrieval of the device with a 35 mm Gooseneck snare. The procedure was partially successful and a resolutive surgical procedure was needed

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Discussion

Risk factors

Predisposing factors for the device embolization may be device too small, ASA, thick septum secundum (>10 mm), long tunnel^[5] (≥8 mm),^[6] deficient rims of surrounding tissue, and device mispositioning.^[3] In this case, we believe that the choice of a device too small, the presence of an ASA, and a long tunnel could have favored the device leaving from its site. The PFO tunnel was longer than measured at the TOE during the previous deployment: the possible tunnel length underestimation could have led to the choice of an unsuitable device for the PFO characteristics and therefore to its displacement. The patient denied having performed maneuvers potentially able to facilitate the device displacement such as having coughed heavily or repeatedly.^[7],[8] A peculiarity of this case is the finding of this complication in the presence of other congenital defects (BAV and previous AC). The presence of multiple congenital defects may have predisposed the device displacement precluding an adequate device fixation^[9] because of the proximity between the abnormal interatrial septum and the abnormal anatomical features of the proximal ascending aorta (e.g., ascending aorta dilatation); furthermore, the surgical repair of the AC may have favored a more distal drop of the device. The device embolization in the presence of multiple congenital defects represents a unique event in the literature, which gives thought to other possible mechanisms of the PFO occluder device displacement in patients with multiple congenital defects.

Symptoms

The mechanisms underlying the symptoms remain unclear. We assume that the onset of dyspnea after the intervention was caused by the worsening of the right–left shunt (provoked by exercise) due to the PFO enlargement following the device displacement; this may be supported by the detection of a significant oxygen desaturation on the 6-min walking test (98% to 92% after 2 min), an event already observed in other patients with PFO, defined as provoked exercise desaturation.^[10] We excluded platypnea-orthodeoxia syndrome, as the patient had a normal arterial oxygen saturation in a standing position. We also presume that the chest pain radiated to the limbs, after having excluded statin-induced myopathy (normal creatine kinase), was the expression of an aortic syndrome with atypical features, due to the mechanical stress of the foreign body into the abdominal aorta. This may be confirmed by its disappearance after the device retrieval.

Follow-up

Finally, we believe that if a TTE before discharge or a TTE with bubble study within 1 month from discharge had been performed, the problem would certainly have been identified earlier and with lower risk for the patient. Indeed, the current guidelines recommend TTE before discharge, TTE with bubble study after 1, 6, and 12 months and then, every 1–2 years.^[3] However, a correct follow-up probably was hindered by the coronavirus disease-19 pandemic.

Conclusion

As shown, the embolization of a PFO occluder device is a very rare but possible complication (in this case reported for the first time in a patient with multiple congenital defects). For this reason, we strongly recommend paying attention to (1) risk factors before the intervention (PFO characteristics and multiple congenital defects) to correctly choose the device to be placed, and to (2) the clinical-echocardiographic follow-up to recognize and treat this severe complication as soon as possible.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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