Abstract
Case Summary
We present a case of a 31-year-old man with a history of aortic valve endocarditis and surgical aortic intervention. Computed tomography revealed a complex serpiginous fistula with 2 openings between the left ventricular outflow tract and the left atrium. Using 3-dimensional printing simulation for device fit testing and planning, the “mother-in-daughter” system, multimodality guidance with computed tomography angiography C-arm prediction, and 3-dimensional transesophageal echocardiogram guidance successfully guided an 18-mm Cribriform Amplatzer device deployed via a retrograde approach.
Take-Home Message
Three-dimensional printing simulation significantly enhances preprocedural planning and device selection for this complex fistula closure, facilitating successful percutaneous repairs in high-risk patients.
Key Words: 3D printing, CTA, fistula, LA, LVOT, TEE
Graphical Abstract

A 31-year-old man with a history of aortic valve (AV) endocarditis was treated surgically with AV replacement (23-mm Inspiris), mitral repair with the hemi-commando procedure, and aortic root abscess debridement. A 1-month follow-up echocardiogram revealed abnormal color Doppler flow into the left atrium (LA); a follow-up transesophageal echocardiogram (TEE) demonstrated a fistula between the left ventricular outflow tract (LVOT) and LA (Figure 1B). There was normal function of aortic bioprosthesis and mitral leaflets. A gated computed tomography angiogram (CTA) showed the defect to be 20.1 mm long, and 18.7 × 11 mm in diameters on the LVOT side and 6.1 × 9 mm in diameters on the LA side (Figures 1A and 1C). The tract appeared to be complex in view of a serpiginous tract with 2 openings of different sizes. Given high re-do surgical risk, the multidisciplinary heart team recommended to first attempt a percutaneous repair. To plan the procedure, a 3-dimensional (3D) print of his heart was used to fit test a series of occluder devices. Three-dimensional printing, although not without its caveats, allowed for fit testing multiple device sizes and types in rapid succession. Ultimately, it served as an effective visual communication device that facilitated discussion and planning between the imaging team and interventional cardiology team. An 18-mm Cribriform Amplatzer (Abbott) seemed to fit the defect length and diameters best (Figure 1D) compared with ventricular septal defect and Amplatzer septal occluder devices. We simulated the procedure using both antegrade and retrograde approaches with the 3D printed model. The fistula was adjacent to a significant portion of the thin fossa ovalis, and there was a high risk of perforating the fistula from the left atrial side, creating an additional communication. The extreme acute angulation of the interatrial septum and defect would potentially make the antegrade approach more challenging and provide inadequate support to deliver the device into the fistula. The retrograde approach based on our modeling was considered to be more likely to achieve success in a complex case.
Take-Home Messages
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This case highlights the importance of a planned 3D printed simulation to choose the best appropriate device for these fistula closures.
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Achieving entry into the fistula necessitated the combination use of the 3D simulation, “mother-in-daughter” system, computed tomography angiogram C-arm prediction, and 3D transesophageal echocardiogram guidance to enhance procedural success.
Figure 1.
3D Printing Simulation and Approach of LVOT to LA Fistula Closure
(A) CT showing the fistula communication between the LVOT and LA (yellow line and arrow). (B) TEE demonstrating the fistula tract from the LVOT to LA (white arrow). (C) CT 3D image showing the size of the fistula defect (yellow arrow). (D) 3D printing model showing the fistula tract (yellow arrow) and simulated with different devices. (E) “Mother-in-daughter” crossing the fistula. (F) TEE showing BMW across the fistula. (G) IM catheter telescoped into the LA. (H) TEE showing the IM catheter in the LA via the fistula. (I) Destination sheath across the fistula over an Extra Stiff wire. (J) Exposing a cribriform disc in the LA and then pulled back to oppose the LA side as shown in the orange arrow. (K) Cribriform deployed, with the LVOT disc deployed without interaction with the aortic valve. (L) TEE and 3D images showing a cribriform deployed opposing the LA and LVOT sides and no obvious flow across the device. (M) TEE 3D images illustrating the relationship between the device and the aortic and mitral side. 3D = 3-dimensional; AMVL = anterior mitral valve leaflet; AV = aortic valve; CT = computed tomography; LA = left atrium; LV = left ventricle; LVOT = left ventricular outflow tract; RA = right atrium; TEE = transesophageal echocardiogram.
Access was gained via the right femoral artery using a 10-F sheath. A “mother-in-daughter” system was used, consisting of an 8-F JR 4.0 guiding catheter paired with a 5-F, 125-cm IM diagnostic catheter, delivered retrogradely across AV to engage the fistula under TEE and fluoroscopic guidance. CTA C-arm angulation prediction was used to aid crossing. A 300-cm BMW wire (Abbott) was used to cross the fistula into the LA (Figures 1E and 1F, Videos 1 and 2). The wire was advanced under TEE guidance into the pulmonary vein, which allowed enough wire support to telescope the IM catheter into the LA (Figures 1G and 1H).
An Amplatz Extra Stiff wire (Abbott) was advanced into the LA, over which a 7-F destination sheath (Terumo) was advanced (Figure 1I). The 18-mm Cribriform device was then deployed safely, with one disc in the LA and the other in the LVOT, resulting in only trivial flow through the Cribriform (Figures 1J to 1M, Video 3, Video 4, Video 5). There were also no interactions between the device and the aortic or mitral valves, and there was no significant increase in LVOT gradient. Next day and 30-day follow-up TTE showed no residual flow through the fistula.
There are no devices engineered specifically for percutaneous repair of these types of fistulas. The upfront 3D printing simulation, the “mother-in-daughter” system, multimodality guidance with CTA C-arm prediction, and 3D TEE guidance were crucial for entry into the fistula.
Funding Support and Author Disclosures
Dr B. P. O'Neill is a consultant to and receives research support from Edwards Lifesciences. Dr Frisoli is a proctor for Edwards Lifesciences, Abbott, Boston Scientific, and Medtronic. Dr Lee is a consultant for Edwards Lifesciences and proctor for Abbott. Dr Villablanca is a consultant for Edwards Lifesciences, Medtronic, Shockwave, Abiomed, and Angiodynamics. All other authors have reported that they have no relationships relevant to the contents of this paper to disclose.
Footnotes
The authors attest they are in compliance with human studies committees and animal welfare regulations of the authors’ institutions and Food and Drug Administration guidelines, including patient consent where appropriate. For more information, visit the Author Center.
For supplemental videos, please see the online version of this paper.
Contributor Information
Leo Kar Lok Lai, Email: l.k.l.lai816@outlook.com.
Tiberio M. Frisoli, Email: tfrisol1@hfhs.org.
Appendix
With the “Mother-in-Daughter” System, a BMW Wire Used to Cross the Fistula Successfully
Transesophageal Echocardiogram Showing the Success of Wiring the Fistula Using the “Mother-in-Daughter” System
Deployment of the 18-mm Cribriform on the Left Ventricular Outflow Tract Side
Three-Dimensional Transesophageal Echocardiogram Showing the Deployed 18-mm Cribriform Across the Fistula
Video Showing Trivial Contrast Across the Fistula After Deployment of the Cribriform Device
Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Supplementary Materials
With the “Mother-in-Daughter” System, a BMW Wire Used to Cross the Fistula Successfully
Transesophageal Echocardiogram Showing the Success of Wiring the Fistula Using the “Mother-in-Daughter” System
Deployment of the 18-mm Cribriform on the Left Ventricular Outflow Tract Side
Three-Dimensional Transesophageal Echocardiogram Showing the Deployed 18-mm Cribriform Across the Fistula
Video Showing Trivial Contrast Across the Fistula After Deployment of the Cribriform Device

