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Stapling as a method of closure of ductus arteriosus aneurysm in the adult
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     a State University of New York at Buffalo, School of Medicine and Biomedical Sciences, Buffalo, NY, USA

    b Erie County Medical Center, Buffalo, NY, USA

    c Department of Cardiac Surgery, Self Regional Medical Center, Greenwood, SC, USA

    Abstract

    The purpose of this paper is to describe the preoperative diagnosis and the surgical treatment of a symptomatic ductus arteriousus aneurysm in a 65-year-old adult. The diagnosis was made by CT scan of the chest and confirmed by aortic angiography. The aneurysm was stapled and resected uneventfully through a left posteralateral thoracotomy. Early diagnosis and treatment methods were reviewed in the literature; and simple operative interventions were recommended for the future.

    Key Words: Ductus arterious aneurysm in the adult; Treatment: aneurysmorrhaphy

    1. Introduction

    Ductus Arteriosus Aneurysm (DAA) in infancy is a rare congenital anomaly, approximately 0.8% of all malformations. In the adult it is one of the rarest malformations of the aortic arch, approximately 5.2% of all adult thoracic aneurysms [1]. Preoperative diagnosis is difficult, since it usually presents with its late complications: recurrent nerve palsy, obstruction of airways, indentation and erosion to the esophagus, bronchus, pulmonary artery, septic emboli, dissection, etc.

    2. Materials and methods

    We report an adult case with a distal arch aneurysm (DAA) that was diagnosed preoperatively before its usual late complications ensued and was operated upon with stapling of the aneurysm.

    3. Case report

    A 65-year-old male patient with history of carcinoma of the floor of the mouth, resected six years ago. Regular follow-up chest X-rays were negative until this year when left lower lobe atelectasis and minimal amount of pleural effusion was noted. These findings indicated a CT scan with contrast for ‘cancer screening’ which demonstrated ‘the triple star sign’ (Fig. 1) and made it suspicious for an aneurysm of the distal aortic arch. Aortogram confirmed the diagnosis of ductus arteriosus aneurysm (DAA). The angiogram clearly indicated that the sac contains grumous material not quite consistent with the size of the aneurysm at operation.

    He also had another congenital variation: the left vertebral artery originating abnormally directly from the aortic arch between the left carotid and left subclavian arteries (Fig. 2). The aneurysm was exposed through a left thoracotomy. The phrenic nerve, recurrent laryngeal nerve and the vagus nerve were isolated. The aneurysm measured 10–15 cm, the neck was about 5 cm. The stapling was performed by an arterial stapler1 through the neck of the aneurysm, while the blood pressure was temporarily lowered to 30–40 mmHg.

    The aneurysm was excluded from the aorta, opened and resected. Grumous material extruded from inside. There were no leaks through the stapling line. Preparations before the stapling were made for possible aortic clamping and/or cardiopulmonary bypass if needed.

    On pathology the wall of the aneurysm contained ‘fragments of fibromuscular tissue with focal hemorrhage, and peripheral chronic inflammatory cell infiltrate.’

    4. Results

    The patient had an uneventful postoperative course and on follow-up studies, 3 weeks and 2 years later. A reconstructed CT scan documented complete exclusion of the aneurysm and a normal size aorta.

    5. Discussion

    In 1983, Mitchell et al. [2] reported 5 adult cases with an established preoperative diagnosis who were operated upon without mortality; one of the patients had aneurysmorrhapy. This brought up the total adult cases in the literature to 21.

    In 1991, Lund et al. [3] reported altogether 106 cases from the literature, 32 of them were discovered in adulthood.

    In the adult, early presentation may include laryngeal nerve palsy, an abnormal chest X-ray, and/or a lesion located in the aortopulmonary window [4]; while the extreme presentation is often sudden rupture and death (3), obstruction of the esophagus, indentation and pressure on the bronchus and/or on the pulmonary artery etc. as noted by the previous reports [5]. Consequently a clear indication for surgery existed even in our asymptomatic patient.

    Primary DAA may occur in any age. Lund divided his cases in 3 groups: adult, children and infant aneurysms. He collected 32 adult cases in 1991, 9 childhood cases (2 months to 15 years) and 65 infant cases (<2 months).

    The complication rate in infants younger than 2 months was 31%, in children (2 months to 15 years) 66% and in adults 47%. One hundred and six were designated being spontaneous, of these 38 were complicated by previous surgical and invasive treatment. Operative mortality was 25% less than 2 months of age; 0% in children and 7% in the adults.

    This lesion in the adult is potentially dangerous since extremely serious emergency complications can ensue; at that point surgery is done as a last resort [6].

    A consensus has been established in the literature by Cruickshank [7] that the ductus arteriosus aneurysm:

    Should not have an arteriosclerotic process in the area.

    The aneurysm should protrude towards the ductus and/or to the pulmonary artery. (Not a ‘Ductus bump’)

    In the adult the ductus should be occluded forming a closed fibrous strand as it was in our case.

    Unfortunately, due to lack of specific studies we have no proof that our patient's aneurysm already existed in his childhood or if it developed spontaneously [8].

    With high speed spiral CT (triple star sign) and MRA [9] a dilation of the ductus (‘ductus bump’) should be clearly differentiated from an aneurysm. Additional diagnostic tests may include 3D echo [8] and/or angiography.

    Different devices were used to close the PDA during infancy in the presence of DAA. Apparently the application of these devices may be associated with later complications of the aneurysm necessitating reoperative surgery [10]. Before 1983 about equal number of children and adults had attempted resection of DAA with failure.

    Second attempted surgical closure carried the highest mortality (26%) [10]. Without surgical intervention 91% of the patients died due to rupture, infection, etc.

    Various operative techniques have been used throughout the years including cardiopulmonary bypass [2], separate perfusion of the arch and the lower extremities [5], circulatory arrest [8]. In a previous case (by the author J.B. Thomson), a 62-year-old male patient's aneurysm was closed with the help of a Gott shunt by-passing the aneurysm and patching the neck of the aneurysm from inside the aorta [4]. In our case the aneurysm was stapled and resected.

    In cases of a ‘true’ uncomplicated ductus aneurysm having a ‘narrow neck’ the simplest technique to use is by hand sewing aneurysmorrhaphy or stapling as reported above. More elaborate methods of repair have to be applied when complications ensue.

    Footnotes

    1 Auto Suture Roticulator 30, Roticulator 30-V3 and Roticulator%% Disposable Surgical Staplers (United States Surgical Corporation, Norwalk, Connecticut 06856, 1995, United States Surgical Corporation)

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    Mitchell RS, Seifert FC, Miller DC, Jamieson SW, Shumway NE. Aneurysm of the diverticulum of the ductus arteriosus in the adult. J Thorac Cardiovasc Surg 1983;86:400–8.

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    Egami J, Tada Y, Takagi A, Sato O, Idezuki Y. False aneurysm as a late complication of division of a patent ductus arteriosus. Ann Thorac Surg 1992;53:901–2.(Thomas Z. Lajos, John Bel)