Ann Thorac Surg 2000;69:282-283
© 2000 The Society of Thoracic Surgeons
Case Reports
Successful resection of ductus arteriosus aneurysm in infancy
Vidmantas J. Zilinskas, MDa,
Stanislovas Maknavicius, MDa,
Giedrius Baliulis, MDa
a Department of Thoracic Surgery, Vilnius University Children’s Hospital, Vilnius, Lithuania
Address reprint requests to Dr
Zilinskas, Heart Surgery Clinic, University Hospital Santariskes, Santariskiu 2, Vilnius 2021, Lithuania
e-mail: notb{at}taide.lt
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Abstract
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We report a case of thrombosed patent ductus arteriosus aneurysm in an infant. The aneurysm was detected accidentally on chest roentgenogram and presented as globular soft tissue density mass in left posterosuperior mediastinum. Resection of the aneurysm was performed without cardiopulmonary bypass. Two years after operation the patient is well and growing normally.
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Introduction
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Prolonged or persistent patency of the ductus arteriosus (PDA) is a common malformation in neonates and infants. It can occur as an isolated defect or in association with other heart defects. Rarely, the ductus arteriosus can be aneurysmal, although the aneurysm of ductus arteriosus is a rare, but potentially fatal condition [1]. In this report we describe a case of thrombosed PDA aneurysm in an infant which was successfully resected.
A 3.6 kg male infant developed the symptoms of respiratory distress on the fifth day of life and was admitted to the Children’s Hospital of Vilnius University. A chest roentgenogram showed a 2 to 2.5-cm globular mass in the left posterosuperior mediastinum. Physical examination revealed systolodiastolic heart murmur. Two-dimensional echocardiography demonstrated patent ductus arteriosus (PDA) (inner diameter 0.25 cm) with large left to right shunt. A computed tomographic scan showed a globular soft tissue density mass in the left posterosuperior mediastinum closely associated with vertebral column and cardiac contours (Fig 1). A thrombosed aneurysm of PDA and neurogenic tumor were considered in the differential diagnosis for the mediastinal mass. Left lateral thoracotomy for the mediastinal mass confirmed the diagnosis of the thrombosed PDA aneurysm. The aneurysmal dilatation was found to involve the aortic end of the PDA and the anterior wall of the descending aorta. It was decided that the operation would be performed without cardiopulmonary bypass. The aneurysm, descending aorta, left subclavian artery, and the pulmonary end PDA were dissected free. While performing dissection, the patient’s core temperature was cooled down to 33°C. The patient was heparinized (50 IU/kg). Resection of the aneurysm was performed by placing vascular clamps on the descending aorta proximally and distally to the aneurysm and on the pulmonary part of PDA. The lumen of the aneurysm appeared to be fully thrombosed. After the resection of the aneurysm, a defect remained on the wall of the aorta, which was closed with a patch of autologous pericardium. The pulmonary stump of PDA was oversewn with 6-0 Prolene (Ethicon, Sommerville, NJ). Aorta cross-clamping time was 18 minutes. The patient had an uneventful postoperative recovery, and was well and growing normally at the 2 year postoperative follow-up examination. A computed tomographic scan also did not reveal abnormal findings (Fig 2).
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Fig 2. Computed tomographic scan of the chest 2 years after operation. The descending aorta is of normal diameter.
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Comment
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Aneurysmal dilatation of the ductus arteriosus is a very rare lesion found in both infants and adults. The aneurysms are classified as spontaneous and acquired, the latter developing as a complication after surgical repair of PDA. The spontaneous aneurysmal dilatation of PDA is met at any age, but is most commonly found in children under 2 months of age [1]. The pathogenesis of PDA aneurysm remains unknown. Two different theories have been proposed concerning the mechanism for spontaneous infantile dilatation of the ductus arteriosus.
The theory proposed by Dr Helen Taussig in 1947, suggests that the primary event in aneurysm formation is failure of the duct to close at the aortic end after the pulmonary end has closed [1, 2]. Physiologically, the closure of PDA begins at the pulmonary end of the ductus. Simultaneously, ductal tissue undergoes involutional changes that result in a decrease in elastic tissue and increase in fibrous tissue, resulting in a weakened wall. If the aortic end of the ductus fails to close, the weakened wall of the ductus, subjected to systemic arterial pressure, may undergo aneurysmal dilatation. The theory is supported by the fact that about 30% of infant PDA aneurysms were closed at the pulmonary end while the aortic end was always patent [1].
The second theory suggests that the congenital or acquired structural abnormality of the wall of the aorta near the ductus to be responsible for the aneurysmal dilatation of PDA. Some authors have reported histological changes of the PDA aneurysm wall [3, 4]. Our case supports the latter theory, as the pulmonary end of PDA was patent and the histology of the aneurysm wall revealed areas of degeneration in the media of the vessel, although there was no evidence of connective tissue disease in our patient. Recently Sven Laurin reported PDA aneurysm with calcified wall present at birth, pointing to a congenital origin of the aneurysm [5].
Because of its rarity, PDA aneurysm may present some difficulties in diagnosis. While transthoracic echocardiography and angiography are an established approach to confirm the diagnosis of nonthrombosed PDA aneurysm [3, 4, 6], the diagnosis may be complicated when PDA aneurysm is thrombosed [2, 7]. In such cases, the final diagnosis may be confirmed only on the thoracotomy for the mediastinal mass, as it was in our case. Although it has been reported that angiography can be helpful, even when the ductus aneurysm is partially filled with clot [2], we decided not to perform angiography because the operation had to be performed for the mediastinal mass, whatever the diagnosis.
PDA aneurysms are associated with potentially serious complications, such as rupture, erosion, infection, and thromboembolism. The rate of complications in infants younger than 2 months of age was reported to be 31% [1]. Therefore, operation should be recommended for infantile ducts arteriosus aneurysm if it does not regress spontaneously. The operation could potentially be performed without bypass, with cardiopulmonary bypass, or alternatively, with hypothermic circulatory arrest. In our case, this procedure was performed without cardiopulmonary bypass, as it took place in a hospital with no facilities for cardiopulmonary bypass, however we believe that operation without cardiopulmonary bypass may be the procedure of choice in cases of favorable anatomy. The use of autologous pericardium was also dictated by lack of cardiac surgical prosthetic materials.
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References
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D’Udekem Y., Rubay J.E., Sluysman T. A case of neonatal ductus arteriosus aneurysm. Cardiovasc Surg 1997;5:338-339.[Medline]
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Sattar P., Ehrensperger J., Ducommun J.C. Thrombosed aneurysmal nonpatent ductus arteriosus. Pediatr Radiol 1996;26:207-209.[Medline]
Accepted for publication June 2, 1999.
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Abstract
Introduction
