Cases reported "Hypertension, Pulmonary"

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1/9. Single lung transplantation for primary pulmonary hypertension without cardiopulmonary bypass.

    We report the first case, to our knowledge, of single lung transplantation for primary pulmonary hypertension carried out without cardiopulmonary bypass. This operation seems to be possible even if the right ventricular ejection fraction is low (0.17) and the pulmonary vascular resistance very high (1,096 dynes.s.cm5). Since 1981, heart-lung transplantation has been successfully performed in patients with primary pulmonary hypertension. If heart-lung transplantation results in resolution of pulmonary hypertension, the incidence of obliterative bronchiolitis is significant in heart-lung transplantation recipients. Single lung transplantation has been performed for end-stage interstitial and obstructive lung disease but has not been considered a good option for primary pulmonary hypertension due to concerns that a single transplanted lung would be unable to cope with the entire blood flow. However, recently single lung transplantation has been performed for primary pulmonary hypertension, the risk of obliterative bronchiolitis remaining unknown. The purpose of this communication is to report one case of single lung transplantation for primary pulmonary hypertension and the feasibility of this operation without the use of cardiopulmonary bypass, if cardiopulmonary bypass is thought to be dangerous.
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2/9. Spontaneous echo contrast of unexpected etiology.

    cor triatriatum sinister is a rare congenital anomaly characterized by the presence of a fibromuscular membrane dividing the left atrium into two chambers: one entering the four pulmonary veins, the other connecting to the mitral valve. The extent of the communication between the two chambers and the presence of associated lesions determine the severity of symptoms and the complications. We report the case of a 20-year-old man firstly diagnosed with obstructive cor triatriatum sinister and severe pulmonary hypertension.
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3/9. Treatment of a case with right-sided diaphragmatic hernia associated with an abnormal vessel communication between a herniated liver and the right lung.

    We herein report a rare case of a newborn girl with a right-sided congenital diaphragmatic hernia where a herniated liver had an abnormal vessel communication with the right lung. A surgical repair was initially attempted through a thoracic approach at 4 days of age where only a plication of the hernia capsule was performed without a prominent improvement in the respiratory condition. At 1 year of age, an angiographic examination was performed, which revealed pulmonary hypertension and abnormal vessel communications where the right pulmonary flow returned to the herniated liver and the right lung also received an arterial supply from the liver. During the second surgical repair performed at 1 year and 1 month of age, an abdominal approach through a right subcostal incision was selected. The aberrant vessels between the lung and the liver were carefully identified and ligated. Because the right lobe of the liver was completely herniated, a hepatic segmentectomy of S6 and S7 was performed. The patient has been doing well for 21 months without any mechanical ventilation since 2 months after undergoing the radical second operation. When performing surgery on a right-sided diaphragmatic hernia, the potential presence of such a vessel anomaly should be carefully taken into consideration.
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4/9. hypoplastic left heart syndrome with intact atrial septum--attempt of an interventional palliation by ductal and interatrial stent implantation.

    In patients with hypoplastic left heart syndrome (HLHS) and intact atrial septum, the blood entering the left atrium cannot egress. emergency treatment interventionally or surgically is mandatory immediately after birth. We describe a patient with HLHS and intact atrial septum who underwent successful transvenous atrial septostomy immediately after birth. When the interatrial communication became restrictive, stent implantation into the arterial duct and into the atrial septum was performed on the 7th day of life. Despite good hemodymanic response, the lung damage was severe and persistent, rendering staged surgical correction impossible. The child died on the 23rd day of life. autopsy showed patent and correct placed stents in the duct and the atrial septum. There was severe dilatation of pulmonary lymphatic and venous vessels, suggestive of long-standing pulmonary venous hypertension. In conclusion, this form of HLHS has a poor prognosis despite early and aggressive interventional treatment.
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5/9. Severe pulmonary hypertension and multiple left coronary arterial fistulas in association with congenital hepatic fibrosis.

    Multiple fistulous communications between the left anterior descending coronary artery and the left ventricle were found in a 55 year old woman with congenital hepatic fibrosis presenting with breathlessness. At cardiac catheterisation severe pulmonary hypertension was also found. In view of the persistent hypoprothrombinaemia, severe thrombocytopenia, and the multiple fistulas the risk of operation was thought to be unacceptable and she continues on medical treatment.
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6/9. Double outlet left ventricle (report of a case with intraventricular surgical repair).

    A case of successful surgical treatment of double outlet left ventricle is reported. In a 7 year-old patient with this rare congenital cardiac malformation, situs solitus of the viscera and atria and concordant connexion of atria and ventricles, d-malformation of the great arteries with the aorta to the right and slightly posterior to the pulmonary artery, large doubly committed ventricular septal defect and neither a subpulmonary nor subaortic infundibulum, the repair was achieved performing intraventricular rerouting of the blood from the right ventricle to the pulmonary artery and closing the interventricular communication with a synthetic patch.
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7/9. Iliac arteriovenous fistula due to spinal disk surgery. Causes severe hemodynamic repercussion with pulmonary hypertension.

    We present a case of a 46-year-old man with a pulsatile mass in the left inferior abdominal quadrant that irradiated a continuous murmur extending to the left lumbar region. Despite an 8-year history of cardiomegaly, he appeared to be asymptomatic except for the mass and could recollect no traumatic injury or surgery that might have caused it. Near the vertebral column, we found a small scar, the result of spinal disk surgery 11 years before. Following chest radiography and electrocardiography, we located the suspected arteriovenous fistula by selective angiography of the aorta and its branches: a communication of the left iliac artery with the left iliac vein had resulted in a very large left-to-right shunt and a severely dilated inferior vena cava. We then divided and isolated the arterial segment containing the fistula, but left this segment in continuity with the left iliac vein by over-sewing both ends. To avoid injury to surrounding structures, dissection was limited to the area of maximal thrill. Hemodynamic improvement was immediate, and the postoperative course was uneventful. At the present time, almost 3 years postoperatively, the patient is asymptomatic.
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8/9. Oxygenation improvement with nitric oxide in right-to-left shunt without significant effects on pulmonary arterial pressure.

    Following surgical closure of an interventricular communication complicating an anterior myocardial infarction, a 74-year-old woman developed severe right ventricular failure and hypoxemia due to the opening of a patent foramen ovale (PFO). Mean pulmonary artery pressure was 24 mm Hg. Treatment with inhaled nitric oxide (5 ppm) increased PaO2 from 47 to 90 mm Hg (FIo(2)1). The present observation points out that nitric oxide inhalation could be useful in the management of severe hypoxemia from a right-to-left shunt due to a PFO even when there is no significant pulmonary hypertension present.
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9/9. Non-cirrhotic portal fibrosis associated with pulmonary arteriovenous communication and pulmonary arterial hypertension.

    A case of non-cirrhotic portal fibrosis associated with pulmonary arteriovenous communication and pulmonary arterial hypertension is reported. The patient was a 7-year old boy who presented with hematemesis, cyanosis, hypoxemia and orthodeoxia. His liver pathology was compatible with non-cirrhotic portal fibrosis. His pulmonary angiography showed arteriovenous shunting and pulmonary arterial hypertension (mean pulmonary artery pressure 34 mmHg). His sister also had non-cirrhotic portal fibrosis with neither hypoxemia nor orthodeoxia. This report raises the possibility of non-cirrhotic portal fibrosis having a genetic etiology.
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