Core Tip: Patients who suffer from atrial septal defect (ASD) with atrial fibrillation are prone to right heart dysfunction and embolism. We report the first case treated with a 3-in-1 procedure (cryoballoon pulmonary vein isolation and left atrial appendage occlusion prior to ASD closure), which may not be performed routinely. However, for ASD patients complicated with poorly controlled atrial fibrillation and unable to tolerate long-term oral anticoagulants, this 3-in-1 procedure can be considered.
INTRODUCTION
Pulmonary vein isolation (PVI) has been established as a treatment for patients with atrial fibrillation (AF)[1]. Cryoballoon PVI has become a relatively simple alternative for radiofrequency ablation[2]. Left atrial appendage (LAA) occlusion is performed as an alternative treatment to oral anticoagulation in patients with non-valvular AF[3]. Atrial septal defect (ASD), as the most common congenital heart disease, may lead to right heart dysfunction and paradoxical embolism[4]. In patients who suffer from both AF and ASD, cryoballoon PVI combined with ASD closure and the LAA occlusion combined with ASD closure have been reported[5-7]. However, there has been no report on the 3-in-1 procedure (cryoballoon PVI, LAA occlusion and ASD closure), which may be effective for preventing stroke and right heart failure. Here, we report a patient who underwent sequential cryoballoon PVI, LAA occlusion and ASD closure during the same operation.
CASE PRESENTATION
Chief complaints
A 65-year-old man was admitted to our hospital due to recurrent episodes of palpitations and shortness of breath for 2 years.
History of present illness
His symptoms started 2 years ago with recurrent episodes of palpitations and shortness of breath, which had worsened over the last 48 h.
History of past illness
His past illness included AF, ASD, coronary heart disease with stent implantation and diabetes.
Personal and family history
None.
Physical examination
The patient’s temperature was 36.6 °C, heart rate was 74 bpm, respiratory rate was 16 breaths per minute, blood pressure was 120/70 mmHg and oxygen saturation in room air was 98%. There was no filling of jugular vein; cardiac auscultation showed arrhythmia and no cardiac murmur in each valve area; and no edema was found in both lower limbs.
Laboratory examinations
On admission, his blood tests including routine blood test, renal function, liver function, thyroid function and coagulation function showed no abnormalities.
Imaging examinations
Pulmonary vein (PV) anatomy was assessed in detail by cardiac computed tomography (CT) (Figure 1A). Reconstruction and measurement of LAA and selection of suitable implantation angle and position were also completed by cardiac CT (Figure 1B-C). Electrocardiography showed AF with a ventricular rate of 76 bpm. Echocardiography showed normal left ventricular ejection fraction, moderate dilatation of the left atrium (50 mm), severe enlarged right atrium and right ventricle and moderate tricuspid regurgitation (estimated pulmonary arterial systolic pressure was 47 mmHg). Abnormal flow from the left to right atrium through the interatrial septum was found by color Doppler image. Echocardiography revealed a 25-mm secundum ASD with adequate margins for ASD closure. There were no obvious abnormalities on chest CT and abdominal color Doppler ultrasound.
Figure 1
Reconstruction of pulmonary vein and left atrial appendage by cardiac computed tomography. A: Reconstruction of pulmonary vein; B-C: Reconstruction and measurement of left atrial appendage (LAA); D: LAA occluder was released. Orange arrow shows LAA occluder.
Reconstruction of pulmonary vein and left atrial appendage by cardiac computed tomography. A: Reconstruction of pulmonary vein; B-C: Reconstruction and measurement of left atrial appendage (LAA); D: LAA occluder was released. Orange arrow shows LAA occluder.
Further diagnostic work-up: AF embolism and bleeding score
CHA2DS2VASc score was 3 (diabetes mellitus, vascular disease, age 65 years to 74 years) and HAS-BLED was 1 (age ≥ 65 years). He refused a long-term anti-coagulation treatment.
FINAL DIAGNOSIS
AF, ASD, coronary heart disease and diabetes.
TREATMENT
Cryoballoon PVI
The patient had recurrent AF, which was poorly controlled with antiarrhythmic drugs, so PVI was attempted. Under general anesthesia, a 12F FlexCath steerable sheath (Medtronic Inc., Minneapolis, MN, United States) was advanced into the left atrium without transseptal puncture. A cryoballoon catheter (Medtronic Inc.) was introduced inside the 12F sheath. Following good balloon occlusion, we applied two ablation freezes for 120-180 s (Figure 2A-D). PV mapping was performed following ablation of all four PVs with a 10-pole Lasso catheter (Biosense-Webster Inc., Diamond Bar, CA, United States). We used bidirectional conduction block between the left atrium and PVs[8] to conform the complete elimination of PV electrical activity. Preoperative and postoperative electrocardiograms are shown in Figure 2E-F.
Figure 2
Cryoballoon ablation. A-D: Cryoballoon ablation of all four pulmonary veins with good balloon occlusion; E-F: Preoperative and postoperative electrocardiogram. White arrows show balloon occlusion.
Cryoballoon ablation. A-D: Cryoballoon ablation of all four pulmonary veins with good balloon occlusion; E-F: Preoperative and postoperative electrocardiogram. White arrows show balloon occlusion.
LAA occlusion
Anticoagulant therapy was recommended, but the patient refused to take long-term oral anticoagulants, so LAA occlusion was selected. Following the cryoballoon PVI, the patient underwent LAA occlusion under transesophageal echocardiographic (TEE) monitoring[9]. A special sheathing canal was placed to perform LAA angiography, and a pigtail angiographic catheter was directed to the LAA with the following positions: Right anterior oblique 30° + cranial 20° and right anterior oblique 30° + caudal 20°. Suitable LAA occluder (Watchman, 3.0 cm) was selected following measurement of LAA orifice width and depth. The LAA occluder was introduced into the LAA along the sheathing canal. The position of the occluder was monitored by TEE. A pull test was conducted to determine the stability of the occluder. After suitable position of the occluder and good plugging effect were confirmed, the occluder was released (Figure 1D).
ASD closure
The indications and benefits of atrial septal occlusion are clear. Figure 3A shows a secundum ASD by echocardiography. After cryoballoon PVI and LAA occlusion, the diameter of the interatrial defect was measured on TEE images in various planes, and a 34-mm JIYI ASD occluder device (Shanghai Shape Memory Co., Ltd, Shanghai, China) was implanted[10]. Secured and stable positioning of the occluder was confirmed through a push-pull test (Figure 3B). After unscrewing the occluder from the cable, good positioning of the device was demonstrated by a final TEE examination.
Figure 3
Outcome and follow-up. A: Preoperative echocardiography for atrial septal defect (ASD); B: Final X-ray image after left atrial appendage occlusion and ASD occlusion; C: Postoperative echocardiography for ASD; D: Follow-up at 3 mo by transesophageal echocardiography. Orange arrow shows ASD occluder; white arrow shows left atrial appendage occluder.
Outcome and follow-up. A: Preoperative echocardiography for atrial septal defect (ASD); B: Final X-ray image after left atrial appendage occlusion and ASD occlusion; C: Postoperative echocardiography for ASD; D: Follow-up at 3 mo by transesophageal echocardiography. Orange arrow shows ASD occluder; white arrow shows left atrial appendage occluder.
OUTCOME AND FOLLOW-UP
The patient was subsequently treated with propafenone 150 mg three times a day for 3 mo. Antiplatelet and anticoagulation therapy (clopidogrel and rivaroxaban) was administered following the doctor's advice. Before discharge, correct device positions were confirmed by echocardiography (Figure 3C). A follow-up TEE was performed to confirm proper seating of the devices and to identify thrombi or residual leak at 3 mo (Figure 3D). Both devices were located in proper position, and neither thrombi nor leakage was present. Subsequently, the patient discontinued rivaroxaban and changed to aspirin and clopidogrel.
DISCUSSION
We report an ASD patient with AF who underwent the cryoballoon PVI and LAA occlusion prior to ASD closure, which indicates that this 3-in-1 operation is feasible, but it is not recommended as a routine procedure. For patients with ASD complicated with poorly controlled AF and unable to tolerate long-term oral anticoagulants, this 3-in-1 procedure can be considered.AF is the most common cardiac arrhythmia, which occurs in 1%-2% of the general population[11]. Since PVs were demonstrated as major sources of ectopic beats, PVI has been considered as the cornerstone for AF procedures[12]. Cryoballoon AF ablation has been established as a useful and safe method in treating paroxysmal and persistent AF, providing an alternative approach to radiofrequency ablation[13]. The incidence of AF is strikingly high in patients with ASD, even after surgical closure[14]. Furthermore, compared with the general population, patients with ASD suffer earlier from atrial arrhythmia[15]. Closure of the ASD could decrease the volume overload and reverse remodeling of the atrium[16]. In the present case, we performed cryoballoon ablation followed by closure of LAA and ASD, which we thought could maintain sinus rhythm, reverse atrium remodeling and prevent embolism.Koermendy et al[17] reported that LAA occlusion through ASD or patent foramen ovale was a feasible access. Cardiac tamponade and perforation of adjacent organs could be obviated by avoiding a transseptal puncture[18]. Another advantage is not to create an iatrogenic septal defect. It is not easy to perform LAA occlusion after ASD occlusion, as the ASD occluder makes it difficult to transseptal puncture[19]. Thus, before ASD occlusion, it is necessary to evaluate the indication for LAA occlusion carefully. According to the reported guidelines, a CHA2DS2VASc score of ≥ 2 point is considered as an indication for LAA occlusion[20]. Our case strictly followed this standard, and as this patient refused to take long-term anti-coagulants, LAA occlusion was conducted before ASD closure.Invasive and surgical procedures are becoming less frequent because of the improvement in percutaneous techniques, especially in cardiac interventions[21]. The present case report indicates that cryoballoon PVI and LAA occlusion prior to percutaneous ASD closure can be performed safely and can prevent several difficulties and complications. In addition, this 3-in-1 procedure was beneficial simultaneously to maintain sinus rhythm, reverse atrium remodeling and prevent embolism.
CONCLUSION
Cryoballoon PVI and LAA occlusion prior to ASD closure can be performed sequentially in ASD patients with AF, which may not be performed routinely. However, for ASD patients complicated with poorly controlled AF and unable to tolerate long-term oral anticoagulants, this 3-in-1 procedure can be considered.
ACKNOWLEDGEMENTS
The authors thank Zhang B and Wu DH for their assistance in cardiac CT analysis.
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