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Case History :

A 4-year-old girl with recurrent upper respiratory tract infections was referred for transcatheter closure of a secundum-type ASD at the catheter room. Her total body weight and her height were 14.9 kg and 96 cm respectively.

Clinical Examination:

Notable cardiac examination was reported for a normal S1 and fixed splitting of S2 with a grade III/VI systolic ejection murmur heard at the left upper sternal border. A sinus rhythm with an incomplete right bundle branch block was shown by her electrocardiogram. Mild cardiomegaly with increased pulmonary vascular markings was shown by Chest X-ray.

A secundum ASD measuring approximately 16.2 mm in diameter with left-to right shunting, right atrial and ventricular enlargement, and adequate rims that appeared to be suitable for percutaneous transcatheter closure were demonstrated by transthoracic echocardiography (Fig. 1). A transcatheter ASD closure procedure was attempted with the patient under general anesthesia after a written informed consent was obtained from the patient’s parents. Using a 6F sheath, researchers obtained venous access from the right femoral veins.

At the start of the procedure, administration of an intravenous bolus of weight-adjusted heparin (100 IU/kg) was performed. Mean right atrial pressure of 7 mmHg with ‘‘A’’ wave of 12 mmHg and ‘‘V’’ wave of 10 mmHg and main pulmonary artery pressure of 29/10 mmHg were shown by Cardiac catheterization. A left-to-right shunt was shown at the atrial level by Oximetry with a calculated pulmonary to systemic flow ratio of 1.8:1.  A 10F AGA delivery sheath advanced a 24 mm Amplatzer septal occluder. The left atrial disc was easily prolapsed into the right atrium during deployment of the occluder. Recapture of the device was performed and multiple attempts at deploying the occluder failed. The application of a larger ASO device could interfere with other important intra-cardiac structures as the stretched diameter of the atrial septum was 40 mm.

Moreover, accommodation of the expanded left atrial disc of a larger device would be insufficient with the size of the left atrial cavity. Hence after a total procedure time of 62 min, the procedure was abandoned. Neither aspirin nor heparin after the procedure was administered to the patient. Sudden onset of slurred speech and right-limb movement disorder were noted in the patient on post-operative day 1. Expressive aphasia and right-sided hemiplegic paralysis were found on clinical examination.

Lesions of acute infarction in the leftward hippocampus, basal ganglia, and temporal lobe area were demonstrated by cerebral magnetic resonance imaging (MRI) (Fig. 2). Any cardio-aortic source of embolism was ruled out by performing transthoracic echocardiogram for confirming that all cardiac chambers were clear of thrombus.

No evidence of pulmonary embolism was found on chest computed tomography scan. Extensive venous thrombosis in the right femoral vein color was shown by Doppler of the lower extremities.

Normal protein C, protein S, activated protein C resistance, antithrombin III, homocysteine, and anti-cardiolipin antibody were found on laboratory evaluation.  Responsibilities of the systemic neurological deficits were thought to be due to PDE on the basis of these findings.

The patient was anti-coagulated with titrated doses of Warfarin and commenced aspirin at 50 mg daily. Repeat venous Doppler examination of the right lower extremity performed 15 days later showed no residual thrombus in the right femoral vein. Surgical closure of the ASD was strongly recommended for secondary prevention of recurrent systemic embolization; however, the patient’s parents refused to give consent for the procedure. The patient was then discharged to a rehabilitation facility on oral anticoagulation. She has received intensive physical therapy, but further rehabilitation and recovery has been slow and unfavorable.

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Investigation Reports :

Click on the link below to view the Image/Movie and Description

Discussion :

The term PDE was coined to describe a condition in which emboli of venous system origin enter the systemic arterial circulation by traversing through an abnormal communication between the right and left heart leading to subsequent systemic embolic events.

PDE can be presumed in the presence of the following triad:

  1. Evidence of systemic arterial embolization in the absence of an embolic source in the left heart,
  2. Detection of an embolic source in the venous system, and
  3. Demonstration of an abnormal communication between the venous and arterial systems with a pressure gradient conducive to right-to-left shunting.

The patient in the present case fulfilled all of the three criteria for diagnosis of presumptive PDE.  

Researchers postulated that in this patient an acute physiological increase in right atrial pressure might be expected to occur during coughing or crying that probably caused transient right-to-left shunting, thereby allowing the venous thrombus to enter the left atrium. Then occurrence of systemic neurological sequelae was reported.

One of the major determinants of PDE worth noting as it had occurred in this patient was the presence of femoral deep venous thrombosis (DVT). Clinically, femoral DVT was silent ad when first demonstrated by ultrasonography, lacked signs.

Mechanical factors, including the number of attempts at venipuncture, the number of catheters inserted, the material and diameter of catheter used, and the presence of a hypercoagulable state were associated with the likelihood of developing catheter-related DVT.

Researchers believed that the present case was predisposed to the development of femoral DVT due to several underlying factors:

  1. The patient after the procedure did not receive anticoagulation
  2. The vascular intima above the puncture site might have been injured by the use of a 10F delivery sheath thereby leading to increased risk of local thrombosis
  3. Venous return may have been impeded by a tight pressure dressing on the venous puncture site, thereby leading to a degree of venous stasis in the right leg. The development of DVT may have been facilitated by this abnormal venous flow pattern.
  4. High blood viscosity may have been contributed by a relative volume depletion or dehydration caused by fasting during the periprocedural period.

The present case highlights important clinical considerations:

  1. Clinicians should have a high index of suspicion for PDE in younger individuals presenting with cryptogenic stroke
  2. Femoral DVT is a common complication of cardiac catheterization in children but has been overlooked usually.

Moreover an important embolic source of PDE is femoral DVT; thus, physicians should be aware of the risk of femoral DVT after cardiac catheterization. These above mentioned recommendations may be helpful to prevent or minimize the risk of DVT and subsequent PDE in similar cases.