A case of Marfan’s syndrome with atresia of right coronary artery is reported. A 45-year-old woman, who was diagnosed as Marfan’s syndrome 1 year ago, came to the hospital complaining of acute chest pain. The patient showed arachynodactyly, pectus carinatum, and long and slender extremities. In echocardiography there were severe aortic regurgitation measured grade IV and aortic dilatation of ascending aorta maximally 5.9 cm in diameter. Mitral regurgitation was mild, but there were also moderate left ventricular dilation and moderately decreased ejection fraction of left ventricle. At operation, atresia of right coronary artery was found. We performed Bentall type operation with SJM 27mm valved conduit for left coronary artery, and Piehler’s modification for right coronary artery bypass using 6mm PTFE graft. The atretic portion of right coronary artery from the suspected right coronary ostium to distal coronary flow was about 4 cm in length. The combination of right coronary artery atresia and Marfan’s syndrome is very rare. The author describes the rare case, which is treated with combined technique of Bentall and Piehler modification for reconstruction of coronary circulation.
Pulmonary sequestration is a congenital malformation characterized by an area of embryonic lung tissue that derives its blood supply from an anomalous systemic artery. Two forms recognized: extralobar and intralobar. Extralobar form is a very rare congenital malformation, usually located in the lower chest, and may be found in newborn infants at the time a congenital diaphragmatic hernia is repaired. Large sequestrated segments may be cause acute respiratory distress in the neonate. The condition is asymptomatic in 15 per cent of patients. This report presents two cases of pulmonary sequestration which misdiagnosed a superior mediastinal tumor and a benign lung tumor. First case was 30-year-old male patient and chief complaints were dyspnea, dry cough and right chest pain. Chest X-ray showed a homogenous increased density of smooth margin at the right superior mediastinal area and suggested a benign mediastinal tumor. And so explothoracotomy was made without other special studies. Second case was 28-year-old male patient. One month ago, he had tracheostomy and right closed thoracostomy due to massive hemoptysis and spontaneous hemothorax. Chest X-ray showed a benign cystic lesion at RLL area. At the time of operation, in first case, a mass of adult fist size was placed medial to the right upper lobe and densely adhesive to trachea, SVC and esophagus. Blood supply of the mass was bronchial arteries of trachea and RUL bronchus and drained to SVC and azygos vein through anomalous systemic veins. There was no bronchial communication on Frozen biopsy. In 2nd case, large cystic lesion contained old blood hematoma was located in RLL and anomalous blood vessel from thoracic aorta was drained to posterior segment of RLL. In operation field, intralobar pulmonary sequestration was diagnosed, and RLL lobectomy was carried out.
The experimental study for extracorporeal preservation of the heart-lung preparation by autoperfusion system was performed in 10 dogs. Under intravenous Pentothal endotracheal anesthesia bilateral thoracotomies were performed. A 24F cannula connected to a plastic reservoir bag located 100 cm above the level of the heart was introduced into the aortic arch. Left subclavian, innominate artery, and descending aorta were ligated and divided. Both vena cavae were ligated and divided after the bag was half filled with blood. A 24F catheter inserted into right atrium and connected to the plastic bag in order to keep constant the preload. The thoracic trachea was intubated and the lungs were ventilated. The heart-lung preparations were removed en bloc and floated in a $34^{\circ}C$ bath of Hartmann solution. The preparations were observed for from 2 hours to 8 hours, with the average of 5.2 hours. Hemodynamic and hematologic variables were measured during preharvest and autoperfusion. The pH revealed severe respiratory alkalosis due to very low $PaCO_2$ during autoperfusion ; $PaO_2$ remained constant for 130-140 mmHg; $A-aDO_2$ increased markedly. The static inspiratory pressure [SIP] at late autoperfusion [6hr] increased significantly as compared with at early autoperfusion [2hr]. There was no difference between white blood cell counts from right atrium and those of left atrium. Heart rates remained constant for 110-120/min; cardiac outputs maintained to approximately 0.6L/min; mean aortic pressures, 75 mmHg; mean pulmonary arterial pressures, 15-18 mmHg; mean right atrial pressures, 9-13 mmHg; mean left atrial pressures, 12 mmHg lower than those of right atrium. Serum Na maintained with normal range during autoperfusion; K increased significantly; Ca decreased progressively. Hemoglobin and hematocrit decreased significantly during autoperfusion. The study demonstrated that stable hemodynamics could be maintained throughout the experiment and the preparation of the lung seemed to be inadequate, especially after 3-4 hours, such as high $A-aDO_2$, increased SIP, and scattered atelectasis and edema in their gross appearances.
Aortic arch syndrome is an unusual disease entity characterized by the narrowing or obliteration of major branches of the arch of the aorta regardless of etiology. We have experienced 2 cases. One of them was 22 years old office girl with 3 months history of headache, intermittent syncope and weakness and claudication on left arm especially during her physical exercise. On physical examination, pulseless on left antecubital and radial artery and blood pressure on left arm was inable to check and coldness with weakness were noted on the same side. Aortic angiography reealed 34% narrowing of left subclavian artery as that of right. But both common carotid artery and both axillary arterial patency were relatively good. Through right supraclavicular and left axillary incision, bypass graft with Gore-tex prosthesis (I.D. 6mm, Length 25 cm) was implanted from right subclavian artery on 2cm distal to origin of right common carotid arery to left axillary artery distal to axillary fossa. End to side anastomosis with preservation of left subclavian artery was done. Postoperative state was stable with blood pressure of 110/70 mmHg on left arm and palpable antecubital and radial pulsation. Another one was 41 year old male patient with 8 months history of pain and numbness on right upper arm and shoulder. On admission, right arm blood pressure was 110/80 mmHg, left arm was 160/110 mmHg, but other physical findings had no abnormalities. Angiography revealed segmental narrowing of right axillary artery on the beginning with 2 cm in length. Operative treatment with right wupraclavicular and right axillary incision, bypass graft with great saphenous vein (Length; 15 cm) from right subclavian artery between scalenus anticus and medius to axillary artery at distal end of axillary fossa was done. The authors report two cases of Aortic arch syndrome treated with bypass graft using Autograft or Gore-tex with good result.
Atherosclerosis has more than 60% of the causes of arterial occlusive diseases. The abdominal aorta and lower extremity arteries are the most common sites of occlusion. We have treated surgically 2 cases who had intermittent claudication and were diagnosed as simultaneous aortobifemoral and bilateral femoropopliteal obstruction by angiography, but had ineffective results from medical treatment or angioplasty. Simultaneously aortobifemoral bypass using Hemashield Y graft and bilateral femoropopliteal bypass using autologous greater saphenous vein were done. After operations, the symptom disappeared and there were no specific post-operative complications except abdominal wound dehiscence. In postoperative angiography, we had obtained good patency of bypass graft. We are following up patients through the out patient department without recurrence up to 16 months.
Pulmonary sequestration and congenital cystic adenomatoid malformation are two infrequent congenital pulmonary diseases and the combination of these two entities is rare. We had experienced a 3 week old male patient with a pulmonary mass, who had been sufyerring from tachypnea and chest wall retraction after birth. The pulmonary mass was suspected as a congenital cystic adenomat id malformation by chest CT. And therefore, we performed urgent operation via standard thoracotomy incision. Upon a thoracotomy, there was an extrapleural mass with anomalous blood supply near the posterior diaphragm and multiple cystic lesions in right lower lobe. The two anomalous arteries arising from the thoracic aorta and one vein draining into the azygos vein were ligated. and then the extrapleural mass was removed and a right lower lobectomy was performed. Final histologic diagnosis was congenital cystic adenomatoid malformation associated with extralobar pulmonary sequestration. The patient was discharged without any problem.
A 2-year-old, female, Turkish Angora cat was presented due to hindlimb paralysis with 2-day history of vomiting and depression. Physical examination revealed weak femoral pulse and cyanotic footpads of hind limbs. Chest auscultation, thoracic radiography, and electrocardiogram were no remarkable. Abdominal ultrasonography showed suspected hyperechoic thrombus in abdominal aorta. Underlying causes of the arterial thromboembolism were not found on multiple diagnostic examinations and the cat was diagnosed as idiopathic saddle thromboembolism. Despite 4-week regimen with heparin sodium, aspirin, and diltiazem, the hind limbs were progressively and irreversibly necrotized. Therefore, coxofemoral amputation was performed. After surgery, the clinical condition of this cat returned to normal without further complications.
Recently, we met a 12 year old female patient who suffered from bacterial endocarditis and pericarditis which were complicated by patent ductus arteriosus. She was admitted to our hospital because of dyspnea, fever, headache, and generalized ache for 10 days. The initial diagnosis was bacterial endocarditis and pericarditis complicated by patent ductus arteriosus and congestive heart failure. At first, we tried to treat the patient medically with digitalis, diuretics, and massive antibiotics. On echocardiography large amount of pericardial fluid was accumulated mainly right anterior aspect and also noted a large vegetation at pulmonary valve area. With vigorous medical treatment including repeated pericardiocentesis, the patient showed no improvement. So we decided to perform pericardiectomy for elimination of the most probable septic focus. On operation, we encountered an unpredicted event, the pericardium was thickened, distended, and its surface showed pulsating which meant connecting to systemic circulation. We decided to close the operative wound and reoperate her under cardiopulmonary bypass later. On the next day, we operated her under cardiopulmonary bypass later. On the next day we operated her under cardiopulmonary bypass. The operative findings were ruptured main pulmonary artery about 1.5cm in diameter on its ventral portion, the blood from the ruptured main pulmonary artery was filled up the localized pericardial sac due to previous pericarditis. Through the ruptured main pulmonary artery, we also found 0.5cm diametered patent ductus arteriosus. With the aid of partial cardiopulmonary bypass and inserting 24F ballooned Foley catheter at aorta, pericardiectomy was performed first. After completion of the pericardiectomy, total cardiopulmonary bypass was established. With minimum pump flow [0.3L/min/m2] the PDA was closed with two Teflon-felted 4-0 Prolene interrupted sutures. The ruptured main pulmonary artery was also closed using thickened pericardium with three Teflon-felted 4-0 Prolene interrupted sutures. The operation was successful and postoperative course was uneventful. She was discharged on the 16th POD. We report this case as a very rare secondary complication of bacterial endocarditis complicated by patent ductus arteriosus.
We have experienced 44 cases of coarctation of aorta in the age of infancy and children from April 1986 to September 1989 at Seoul National University Children`s hospital. Patients were thirty males and fourteen females, and their age ranged from one month to ten years[mean 23.84 $\pm$33.06 months] with thirty-two infant cases. In the infantile age, congestive heart failure was the most common chief complaint[18/32], and above that age, frequent upper respiratory infection was most common[8/12]. We experienced thirteen cases of isolated COA, twenty-two cases of COA with VSD, eight cases of COA with VSD, eight cases of COA with intracardiac complex anomalies and one case of COA with atrial septal defect. The associated intracardiac complex anomalies were three Taussig-Bing type double outlet right ventricle, one single ventricle, one transposition of great arteries, one atrioventricular septal defect, one hypoplastic aortic arch with left heart hypoplasia, and one Tetralogy of Fallot. Operative techniques of COA were twenty-three subclavian flap arterioplasty, 12 resection and end to end anastomosis, eight onlay patch angioplasty, and I direct angioplasty after resection of web. Among the cases with other cardiac anomalies, staged operation was done in twenty-nine patients, and single stage total correction was performed only in three patients. There were seven operative mortality[15.9%], all being in infantile age group, and among fourteen cases associated with large VSD[Qp/Qs>2.0, mean pulmonary arterial pressure>50mmHg], four patients were died, but there was no mortality in patients with small VSD. With above results, we are intended to discuss about the interval between staged operation, the fate of VSD after coarctoplasty in case of COA with VSD, causes of death, complications etc.
A 56 years old male patient adklitted to our neurology department because of repeated tingling sensation in right 3, 4, 5th. (infers and weakness on grasping, which were progressively developed re ently. At this time, he had also suffered from claudication in both lower extremities. Carotid angiogram showed that right internal carotid artery was obstructed completely, and both common, both external and left internal carotid arteries had significant stenosis, Concommitantly, aortogram suggested complete obstruction just below the renal arteries. We plamled staged operation for two separated arterial lesions. Both carotid endarterectomy was performed. and we used carotid shunt for left side during operation. Abdominal aortic lesion was operated 2 weeks later We obligately clamped aorta just below the celiac artery and infuse4 kidney perservation solution to pertect kidney during ischemia. Reversed Y bypass graft and kidney perservation was successful despite of 40 minute ischemia. Postoperative courts was uneventful and patient was discharged without any specific problem.
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