CASE REPORTS. False Aneurysm After Ligation of a Patent Ductus Arteriosus

Transcription

1 CASE REPORTS False Aneurysm After Ligation of a Patent Ductus Arteriosus Jens G. Rosenkrantz, M.D., Leslie L. Kelminson, M.D., Bruce C. Paton, M.R.C.P., F.R.C.S., and John H. K. Vogel, M.D. T e development of an aneurysm postoperatively has been recognized for many years as a rare complication of surgery for patent ductus arteriosus [l, 2, Surgical treatment is hazardous because of the danger of rupture of the friable, ill-defined aneurysmal wall. This paper describes a relatively safe technique for the excision of this type of aneurysm. The patient (CGH# ), an 8-year-old girl, was first seen at the University of Colorado Medical Center in December, 1965, because of a patent ductus arteriosus. There were no symptoms of cardiac disease, and the murmur, first noted three years previously, was loud, continuous, and heard best at the second left interspace along the lateral sternal border. Peripheral pulses were moderately increased in amplitude, and electrocardiogram and chest x-rays were consistent with the diagnosis of patent ductus arteriosus. She was admitted to the hospital in March, 1966, for operation. Preoperatively she was afebrile, there was no evidence of local or systemic bacterial infection, and her throat culture grew normal flora with many colonies of alphahemolytic stre tococci. The white blood count was 9,500 per cu. mm., with 49% neutrophiles, 14% lymphocytes, 1% monocytes, and 6% eosinophiles. On March 15, 1966, operation was performed, and the ductus arteriosus was found to be 7 mm. in diameter and 10 mm. in length. The pulmonary and aortic ends of the ductus were ligated with cotton umbilical tape, and a transfixion suture of silk was placed between the ligatures. The continuous thrill was eliminated. There was no hemorrhage from the ductus itself. Hemorrhage from many small vessels in the mediastinurn was excessive; operative blood loss was 350 ml., and the From the Departments of Surgery, Pediatrics, and Medicine, University of Colorado Medical Center, Denver, Colo. Accepted for publication Dec. 22, VOL. 3, NO. 4, APRIL, ,

2 ROSENKRANTZ, KELMINSON, PATON, AND VOGEL patient drained 720 ml. of blood from the pleural catheter during the first postoperative day. The drainage then stopped, the pleural space remained empty, and the chest tube was removed on the second postoperative day. During these first two days, the rectal temperature remained about 38.Z C., but after this the patient was afebrile. No antibiotics were administered before, during, or after surgery. On the eighth postoperative day the sutures were removed. There was no murmur. A urine culture had shown a significant number of E. coli, and for this reason she was given ampicillin. Six days later she returned to the hospital because of chills, fever, and cough of two days' duration. A pleural effusion obscured the left lower lung field. Rectal temperature was 4OoC., and the white blood count was 7,300 per cu. mm., with 60% segmented and 5% unsegmented neutrophiles. The pulse rate was rapid, and there was a Grade 2 of 6 low-pitched pansystolic murmur heard best in the second left interspace near the sternal border. No diastolic murmurs were audible. Left thoracentesis yielded 400 ml. of serosanguinous fluid without white cells or bacteria. Moderate widening of the superior mediastinum and an infiltrate throughout the left lung were seen on x-ray. Antibiotic therapy was initiated with intravenous ampicillin, methicillin, and oxycillin. By the following day the physical findings had changed; a continuous murmur had appeared that was loudest over the second left interspace lateral to the sternum and was transmitted over the entire precordium. The peripheral pulse pressure had widened. Beta-hemolytic streptococci were grown from her blood. Penicillin was added to the antibiotic therapy. Within several days the fever resolved, and the atient felt well. The abnormal physical signs persisted. The left pleural e i usion resorbed, but the prominence in the superior mediastinum became more marked. After three weeks of antibiotic therapy and with continued enlargement of the mediastinal mass, cardiac catheterization was performed. A systernic-topulmonary shunt was found at the pulmonary arterial level with a pulmonary-tosystemic blood flow ratio of 1.25 to 1. The mean pulmonary arterial pressure was 37 mm. Hg, and estimated pulmonary resistance was 527 dynes-sec./~m.~ Angiocardiography demonstrated a large aneurysm filling from the arch of the aorta and draining into the pulmonary artery (Fig. 1). On April 21, 1966, 5 weeks after the original operation, reoperation was carried out with total cardiopulmonary bypass. The left femoral artery was exposed. Next, the previous incision in the fourth left intercostal space was opened. This incision was extended anteriorly to the midsternum and then superiorly as a midline sternotomy into the neck. In order that total cardiopulmonary bypass could be started immediately should the aneurysm be entered during the dissection, the pericardium was opened, and venous cannulas were inserted into the right atrium. Two arterial lines, passing through separate pumps, were inserted into the femoral artery and the ascending aorta. FIG. 1. Angiocardiogram, anteroposterior view, showing false aneurysm. 354 THE ANNALS OF THORACIC SURGERY

3 CASE REPORT: Aneurysm After Ligation of Ductus Arteriosus Next, the false aneurysm, which filled almost the entire left upper chest and mediastinum, was dissected off the ascending aorta to a point distal to the left carotid artery. The posterior aspect of the wound was then opened and the descending aorta freed from the adherent aneurysm at the level of the highest pair of intercostal arteries. The left subclavian artery was dissected free high in the mediastinum, yet proximal to its first branches. At this point, cardiopulmonary bypass was instituted. The ascending and descending aorta, the left subclavian artery, and the main pulmonary artery were cross-clamped. The pump flow rates were adjusted so that about one-third of the pump output perfused the ascending aorta; the remainder was directed into the femoral artery. There were no difficulties with perfusion. The false aneurysm was opened and emptied. Its cavity was about 10 cm. in diameter. Its wall was composed of clot and inflammatory tissue, from which dangled the two cotton umbilical tapes and silk transfixion sutures. These ligatures were intact. Smooth openings about 1 cm. in diameter led to the aorta and left pulmonary artery, these vessels making up part of the aneurysmal wall. Although the openings were in the normal location of the ductus arteriosus, no remnant of the du,ctal wall was seen. Each opening was closed with two layers of sutures. The various arterial cross-clamps were then removed, and the aneurysmal wall, intimately adherent to mediastinal structures and left lung, was excised. An unsuccessful attempt was made to identify the left phrenic and vagus nerves. Cardiopulmonary bypass was discontinued after 58 minutes. Blood loss during the procedure was 2,000 ml., most of which arose from the raw surface of the left upper pulmonary lobe. The aneurysmal wall was composed of variably organized granulation tissue with fresh blood clot and pulmonary parenchyma. There were several cleftlike areas of purulent necrosis within the granulation tissue, but no organisms were cultured from the aneurysmal wall or the ligatures. For the first two days after operation, the child was febrile, but later convalescence was uneventful. Postoperative drainage from the chest tubes was minimal. She was maintained on ampicillin, penicillin, and streptomycin postoperatively. The latter two drugs were continued for two weeks and the ampicillin for four weeks. One blood culture on the first postoperative day grew alpha-hemolytic and nonhemolytic streptococci; all subsequent blood cultures were sterile. Cardiac catheterization was repeated 4 weeks postoperatively. There was no evidence of abnormal shunts; the mean pulmonary arterial pressure was 20 mm. Hg, and total pulmonary resistance was 348 dynes.sec./cm.6 Angiocardiogram showed no evidence of aneurysm. The patient was discharged from the hospital and has remained well since. Left phrenic and left recurrent laryngeal palsies have persisted. DISCUSSION In most of the postoperative aneurysms reported, the ductus arteriosus had been ligated; the formation of an aneurysm after division of the ductus has rarely been described. Almost all of these have been false aneurysms, presumably resulting from injury to the ductal wall. Bacterial infection within the aneurysm has been common. Communication may develop with the aorta alone, between aorta and pulmonary artery, or between the aorta and a bronchus [3, 41. In this patient there was probably an unrecognized injury to the ductal wall with accumulation of a hematoma in the mediastinum. The hematoma presumably then became infected and communicated with the aorta and pulmonary artery, forming a mycotic false aneurysm. VOL. 3, NO. 4, APRIL,

4 ROSENKRANTZ, KELMINSON, PATON, AND VOGEL Untreated, these aneurysms have presented the threats of continued infection, rupture with exsanguinating hemorrhage or embolization of clot, vegetation, or even suture material into the pulmonary or systemic circuits [4]. Because of these risks and because of continued enlargement of the aneurysm, this patient was reexplored. The chief hazard of operation has been hemorrhage from the thinwalled aneurysm during dissection, and several patients have died during such hemorrhage. To obtain control of the aneurysm, Ross et al. [61, Hallman and Cooley [l], and Schieppati et al. [7] described cross-clamping of the aorta proximal and distal to the aneurysm and of the left pulmonary artery proximal to its connection with the aneurysm. Coupled with hypothermia, this allowed the surgeon to open the aneurysm and resect its wall without alarming hemorrhage. However, the danger of inadvertently entering the aneurysm during freeing-up of these vessels remains, and this is a particular hazard with large aneurysms. Jones [2] in 1965 reported the use of bypass of the left heart in resecting a postoperative ductal aneurysm, and more recently Morrow and Clark [5] used total cardiopulmonary bypass to permit excision of a large, calcified, spontaneous aneurysm of the ductus. Total cardiopulmonary bypass seemed, in our patient, to offer the safest method of maintaining circulation during removal of the aneurysm because: (1) the femoral artery, ascending aorta, and right atrium could be cannulated without disturbing the aneurysm; (2) had the aneurysm been entered during dissection of the aorta or subclavian artery, the main pulmonary artery could have been clamped, the aorta compressed above and below the aneurysm, and circulation maintained by the pump-oxygenator; and (3) after occlusion of arterial inflow into the aneurysm, the aneurysm could be opened and resected and its communications into aorta and pulmonary artery obliterated without the danger of ischemia of the spinal cord or strain to the left ventricle due to aortic cross-clamping. The period of follow-up is short, and the possibility of recurrent infection or even reestablishment of an aneurysm remains. However, the patient has been afebrile without antibiotics and has remained clinically well. At postoperative cardiac catheterization and angiocardiogram, there was no evidence of recurrent shunt, aneurysm, or mediastinal hematoma. SUMMARY An 8-year-old girl underwent ligation of a patent ductus arteriosus and postoperatively developed a mycotic false aneurysm that communicated with the aorta and pulmonary artery. The aneurysm was excised with the support of total cardiopulmonary bypass. It appears that the 356 THE ANNALS OF THORACIC SURGERY

5 CASE REPORT: Aneurysm After Ligation of Ductus Arteriosus use of total cardiopulmonary bypass affords the safest method of repair of these aneurysms. REFERENCES 1. Hallman, G. L., and Cooley, D. A. False aortic aneurysm following division and suture of a patent ductus arteriosus: Successful excision with hypothermia. J. Cardiov. Surg. 5:23, Jones, J. C. Twenty-five years experience with the surgery of patent ductus arteriosus. J. Thorac. Cardiov. Surg. 50: 149, Jones, J. C. Complications of the surgery of patent ductus arteriosus. J. Thorac. Surg. 16:305, Kerwin, A. J., and Jaffe, F. A. Postoperative aneurysm of the ductus arteriosus. Amer. J. Cardiol. 3:397, Morrow, A. G., and Clark, W. D. Closure of the calcified patent ductus. J. Thorac. Cardiov. Surg. 51:534, Ross, R. S., Feder, F. P., and Spencer, F. C. Aneurysms of the previously ligated patent ductus arteriosus. Circulation 23:350, Schieppati, E., Viola, A. R., and Leyro-Diaz, R. M. Postoperative infected aneurysm of patent ductus arteriosus. Dis. Chest 46:503, Thomas, T., Betts, R. H., Vytilingham, K. I., Gopinath, N., and Saini, V. K. Patent ductus arteriosus. J. Indian Med. Ass. 39: 177, YOL. 3, NO. 4, APRIL,