Caso ClÃnico
Percutaneous resolution of partial anomaly of left pulmonary venous return with dual drainage in pediatric patients. Case report
Rodrigo MartÃn Egües Almeida, Juan AgustÃn EcheverrÃa, Juan Pablo Feldman, Maria Soledad Albisu, Rolando Gómez
Revista Argentina de Cardioangiología Intervencionista 2023;(2): 0071-0072 | Doi: 10.30567/RACI/20232/0071-0072
Introduction. Partial anomalous pulmonary venous return (PAPVR) is a rare type of pulmonary vein anomaly. It is found in 0.5% to 0.7% of the overall population. The presence of a dual drainage system makes a percutaneous approach possible due to the coexistence of both a systemic venous and a normal pulmonary venous circuit.
Objectives.: To report the case report of a patient with PAPVR and dual drainage who was treated percutaneously.
Method:. In this study we present the case report of a patient with PAPVR who was eligible for vertical vein occlusion due to the presence of a dual drainage system.
Conclusions.: Transcatheter device closure of PAPVR with dual drainage is an effective and safe approach.
Palabras clave: dual drainage, partial anomalous pulmonary venous connection, device occlusion.
Introducción. El retorno venoso pulmonar anómalo parcial (APRVP) es una anomalía poco frecuente de las venas pulmonares que se encuentra en el 0,5-0,7% de la población general. La presencia de un doble sistema de drenaje hace posible el abordaje percutáneo debido a la coexistencia de una conexión del circuito venoso sistémico con el circuito venoso pulmonar normal.
Objetivo: reportar el caso de un paciente que presentó anomalía parcial del retorno venoso pulmonar con doble drenaje resuelto percutáneamente.
Método: en este estudio presentamos un caso clínico de un paciente con APRVP, candidato a oclusión de vena vertical por la presencia de un doble sistema de drenaje.
Conclusión. La corrección percutánea con dispositivo de APRVP con doble drenaje es un procedimiento efectivo y seguro.
Keywords: doble drenaje, retorno venoso pulmonar anómalo parcial, dispositivo oclusor.
Los autores declaran no poseer conflictos de intereses.
Fuente de información Colegio Argentino de Cardioangiólogos Intervencionistas. Para solicitudes de reimpresión a Revista Argentina de Cardioangiología intervencionista hacer click aquí.
Recibido 2023-03-25 | Aceptado 2023-05-10 | Publicado
Esta obra está bajo una Licencia Creative Commons Atribución-NoComercial-SinDerivar 4.0 Internacional.
INTRODUCTION
Partial anomalous pulmonary venous return (PAPVR) is a rare anomaly of the pulmonary veins found in 0.5% to 0.7% of the overall population.1 It can have a single (to systemic veins) or dual drainage system (to the systemic atrium and left side).2
Various degrees of right chamber dilation occur due to chronic volume overload due to left-to-right shunting. Increased blood flow to the lungs leads to remodeling of the pulmonary vascular bed triggering pulmonary arterial hypertension.
Management depends on the impact of the left-to-right shunt on heart and lungs, and common treatment includes surgically redirecting the anomalous vein to the left atrium.
The presence of a dual drainage system facilitates the transcatheter approach due to the coexistence of a connection between the systemic venous circuit and the normal pulmonary venous circuit.3,4
OBJECTIVES
To report the case of a patient with partial anomalous pulmonary venous return and dual drainage successfully resolved through a transcatheter procedure.
METHOD
In this study, we present the case of a patient with PAPVR who was eligible for vertical vein occlusion due to the presence of a dual drainage system.
CASE REPORT
This is the case of 13-year-old patient referred to our hospital after being diagnosed with PAPVR of the left superior pulmonary vein. The transthoracic echocardiography performed revealed the presence of PAPVR of the left superior pulmonary vein to a vertical vein with mild dilation of the right atrium, and right ventricle with no signs of pulmonary hypertension.
The coronary computed tomography angiography (CCTA) performed confirmed the presence of PAPVR and a dual drainage system from the left superior pulmonary vein to the left atrium and the innominate vein through a vertical vein (Figure 1A and Figure B).
Cardiac catheterization was performed, and normal pressures were found in the right chambers with a systemic-to-pulmonary shunt (Qp/Qs) ratio of 1.6. There was no pressure gradient between the left pulmonary vein and the left atrium, which is why a decision was made to occlude the vertical vein (Figure 2A).
The vertical vein measured 7 mm, which is why a 14 mm MemoPartTM Plug occluder was selected. Before releasing the device, contrast agent was injected into the left superior pulmonary artery, and no obstruction of the pulmonary vein was seen in the levophase. Complete vessel occlusion without pressure changes and normal drainage in the levophase was confirmed after device release (Figure 2B).
The patient remained hospitalized for 24 hours and underwent echocardiography and x-rays, which showed the proper position of the device and a non-obstructive pattern of venous pulmonary flow. Follow-up at 3 and 6 months after discharge, with echocardiography and CCTA, yielded favorable results.
DISCUSSION
Anomalous pulmonary venous drainage or connection occurs due to 1 or more failed pulmonary veins when connecting to the left atrium during fetal life. PAPVR is most commonly found on the right side and often coexists with a sinus venosus-type of atrial septal defect in 80% to 90% of the cases. Occasionally, the existence of a dual drainage system connecting PAPVR to the left atrium has been reported.2
Diagnosis is often achieved during childhood or adulthood due to the presence of a murmur, arrhythmia or clinical signs of pulmonary hypertension resulting from chronic left-to-right shunting. The “dual drainage” of the left-sided pulmonary veins to both a vertical vein and the left atrium is a condition that should be suspected, and its presence confirmed and diligently assessed on the CCTA and cardiac catheterization, thus allowing a less invasive therapeutic approach.
In a recent series of patients, M.A. Al-Muhaya et al. demonstrated that percutaneous closure with an Amplatzer device for the management of dual drainage PAPVR is an effective, safe, and reproducible approach. It offers many advantages over surgical treatment like fast recovery, avoidance of mechanical ventilation and stay at the PICU, short lengths of stay, low cost, and patient satisfaction.5
Our approach consisted of the percutaneous occlusion of the ascending vertical vein, resulting in the redirection of blood flow to the left atrium with satisfactory results. This is the first report using the MemoPartTM Plug. The MemoPart™ vascular plug is a self-expanding cylindrical device made of a nitinol wire mesh with sewn polyester fabric to enhance occlusion. The device is visible on an x-ray and comes with a controlled release system with a screw-like mechanism similar to the traditional Amplatzer Vascular PlugTM (AVP).
CONCLUSIONS
Percutaneous correction with a dual drainage PAPVR device is an effective and safe procedure.
Allen HD, Driscoll DJ, Shaddy RE, Feltes TF. (2013). Moss & Adams’ Heart Disease in Infants, Children, and Adolescents: Including the Fetus and Young Adult. Lippincott Williams & Wilkins.
Nagulakonda S, Pandey NN, Malhi AS, et al. “Dual Drainage” in the mixed variety of totally anomalous pulmonary venous connection. J Card Surg 2021;1â€2.
Forbess LW, O’Laughlin MP, Harrison JK. Partially anomalous pulmonary venous connection: demonstration of dual drainage allowing nonsurgical correction. Catheterization and Cardiovascular Interventions 1998;44(3):330-5.
Gangadhara MB, Magee AG. Transcatheter occlusion of partial anomalous pulmonary venous connection with dual drainage to left atrium. Ann Pediatr Card 2019;12:144-6.
Al-Muhaya MA, Alkodami AA, Khoshhal S, Najjar AHA, Al-Mutairi M, Abdelrehim AR. Transcatheter occlusion of the vertical vein in a partial anomalous pulmonary venous connection with dual Drainage, case series with literature review. IJC Heart & Vasculature 2021;37:100889.
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Etiquetas
dual drainage, partial anomalous pulmonary venous connection, device occlusion
Tags
doble drenaje, retorno venoso pulmonar anómalo parcial, dispositivo oclusor
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