Unilateral Pulmonary Edema in a Patient with Worsening Tricuspid Valve Regurgitation: A Secret Inside Pulmonary Veins.

We present the case of a 69-year-old patient who was referred to the Department of Echocardiography for surgical treatment of severe tricuspid valve regurgitation (TVR) with advanced congestive heart failure. In 2013 the patient underwent unsuccessful percutaneous ablation for permanent atrial fibrillation. In 2015, following numerous episodes of atrial fibrillation and congestive heart failure with left pleural effusion, the patient was admitted to another center. A transthoracic echocardiogram showed severe TVR and moderate precapillary pulmonary hypertension, confirmed at right cardiac catheterization. He showed bilateral ankle swelling, mild systolic cardiac murmur and localized leftmost decreased breath sounds. Chest X-ray revealed left-sided pulmonary edema and ipsilateral large pleural effusion. Following percutaneous drainage of the left pulmonary effusion, the patient underwent transthoracic and transesophageal echocardiography (TEE), confirming severe TVR due to annular dilation, severe pulmonary hypertension (60 mmHg) and right ventricular overload. At TEE, we found a narrowed single left pulmonary vein. Coronary artery angiography showed no critical stenosis. The patient underwent cardiac magnetic resonance and Angiography that confirmed ostial stenosis of a single left pulmonary vein. We performed successful bare-metal stent implantation. After the procedure, we observed progressive improvement in the patient's clinical condition, concomitant with reverse pulmonary hypertension, significant TVR reduction and chest X-ray normalization. This is a rare case of unilateral pulmonary edema following percutaneous ablation of atrial fibrillation.

INTRODUCTION

Unilateral pulmonary edema following percutaneous ablation of atrial fibrillation is rare. We present the case of a 69-year-old patient who was referred to the Department of Echocardiography for surgical treatment of severe tricuspid valve regurgitation (TVR) with advanced congestive heart failure. His medical history was otherwise unremarkable, apart for moderate TVR due to prolapse. In 2013, the patient underwent unsuccessful percutaneous ablation for permanent atrial fibrillation, with several relapses despite prophylactic antiarrhythmic therapy (flecainide, sotalol). In 2015, following numerous episodes of atrial fibrillation and congestive heart failure with left pleural effusion, the patient was admitted to another center. A transthoracic echocardiogram showed severe TVR, and moderate precapillary pulmonary hypertension was confirmed at right cardiac catheterization.

On arrival at department, the patient was clinically unstable. On initial examination, he showed bilateral ankle swelling, mild systolic cardiac murmur, and localized leftmost decreased breath sounds. Chest X-ray revealed left-sided pulmonary edema and ipsilateral large pleural effusion [Figure 1]. Following percutaneous drainage of the left pulmonary effusion, the patient underwent transthoracic and transesophageal echocardiography (TEE), confirming severe TVR due to annular dilation, severe pulmonary hypertension (60 mmHg), and right ventricular overload. At TEE, we found a narrowed single left pulmonary vein, with increased Doppler flow velocity and color turbulence [Figure 2]. Coronary artery angiography showed no critical stenosis.

Figure 1

Left-sided pulmonary edema and ipsilateral large pleural effusion

Figure 2

Tricuspid insufficient (a) narrowed single left pulmonary vein with increased color turbulence (b). Normal Doppler flow velocity of one right pulmonary vein (c); increasing doppler velocity of single left pulmonary vein (d)

The patient underwent cardiac magnetic resonance showing two pulmonary right veins with regular size and outflow, and a single markedly stenotic superior left pulmonary vein (ostium 4–5 mm) [Figure 3]. Angiography confirmed ostial stenosis of a single left pulmonary vein. We performed successful bare-metal stent implantation [Figure 4].

Figure 3

Single stenotic superior left pulmonary vein (arrow)

Figure 4

Ostial stenosis of a single left pulmonary vein and successful bare-metal stent implantation

After the procedure, we observed progressive improvement in the patient's clinical condition, concomitant with reverse pulmonary hypertension, significant TVR reduction, and chest X-ray normalization [Figure 5].

Figure 5

Chest X-ray normalization after the procedure

This is a rare case of unilateral pulmonary edema in a patient with left pulmonary vein stenosis and TVR-related pulmonary hypertension following percutaneous ablation of atrial fibrillation.

Pulmonary vein stenosis is one of the most serious complications of the percutaneous ablation of atrial fibrillation, occurring in 1% to 3% of cases.[1] Underlying molecular mechanisms remain poorly defined, probably involving scarring, with venous wall contraction and periadventitial inflammation or collagen deposition taking place as a result of thermal injury, which may compromise or even occlude the lumen of the pulmonary vein.[12]

The transcatheter approach is the most common therapy of choice.[3]

TEE is a useful tool to diagnose pulmonary vein stenosis as an injury of percutaneous ablation of atrial fibrillation. Several studies show high diagnosis accuracy for the detection of pulmonary vein stenosis after percutaneous ablation (sensitivity: 82%–100%, specificity 95%–100%) compared to other techniques.[4]

In patients undergoing percutaneous ablation for atrial fibrillation, the occurrence or worsening of TVR, together with progressive pulmonary hypertension, should be carefully evaluated to identify pulmonary vein disease. In our case, with the unusual finding of unilateral pulmonary edema and progressive TVR, TEE was essential to reach an ultimate diagnosis and plan appropriate therapy, avoiding potentially dangerous TVR surgery due to the omission of treatment for unexpected pulmonary vein stenosis.

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Conflicts of interest

There are no conflicts of interest.