Asian Cardiovasc Thorac Ann 2005;13:277-279
© 2005 Asia Publishing EXchange Ltd
Aortic Coarctation Repair with Patch Graft Aortoplasty and Severe Hemoptysis
Kirkpatrick Santo, FRCS,
Alan J Bryan, FRCS,
Raimondo Ascione, FRCS
Bristol Royal Infirmary, Bristol, United Kingdom
For reprint information contact: Raimondo Ascione, FRCS Tel: 44 117 928 3145 Fax: 44 117 929 9737 Email: R.Ascione{at}bristol.ac.uk, Cardiothoracic Surgery, Bristol Royal Infirmary, Bristol BS2 8HW, United Kingdom.
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ABSTRACT
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This report describes the successful treatment of a confirmed case of hemoptysis from a false aneurysm at the site of a previous coarctation repair. Professionals involved in the clinical care of patients that have undergone previous coarctation repair with patch graft aortoplasty, should be aware of the late risk of false aneurysm occurrence. Periodically screening patients with magnetic resonance imaging to prevent potentially fatal complications is recommended.
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INTRODUCTION
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Occurrence of false aneurysm following patch graft aortoplasty is high, and clinically presents with patients suffering conditions ranging from asymptomatic to sudden death due to rupture.1–3 Here, we report a case of a relatively young patient with a previous history of aortic coarctation repair with a patch graft aortoplasty that presented with severe hemoptysis.
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CASE REPORT
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A 39-year old male patient was referred to us with a recent history of hemoptysis, having suffered two severe episodes over a two-week period, each associated with a blood loss of about 1 liter. Past surgical history included a ligation of an arterial duct at an infant age, resection of sub aortic diaphragm at the age of 15, and a repair of aortic coarctation with a Dacron patch graft aortoplasty at the age of 19. The patient had since not suffered from any other significant cardiovascular problems.
Initial investigations with magnetic resonance imaging (MRI) of pharynx and sinuses and an oesophagous-gastro-duodenoscopy, performed prior to referral, had proved normal. The patient’s blood pressure was 130/80, with a heart rate of 68 beats·min–1, normal brachial pulses with no parasternal heave. Cardiac auscultation revealed a gradient 3/6 systolic murmur radiating to the carotids/subclavian region. Clotting screen was unremarkable. A chest radiograph showed an irregular mass adjacent to the aortic knuckle, which was subsequently confirmed to be an enlarged descending thoracic aorta by computed tomography (CT) scan, suggestive of either false aneurysm, or dissection. Due to a previous history of allergic reaction to contrast dye, a MRI was then performed, which showed a large saccular aneurysm of the aorta at the level of the patch graft measuring 6 cm in diameter (Figure 1
and 2). The aneurysm was causing displacement of the trachea, left main bronchus and left pulmonary artery. A supplemental left-sided superior vena cava (SVC) was also observed.
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Figure 2. Transverse section of the aneurysm causing displacement of the trachea, left main bronchus and left pulmonary artery.
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The patient was taken to the operating room and a left thoracotomy was opened through the bed of the 4th rib. The lung was adherent to the chest wall and was carefully mobilized. The femoral vessels were exposed and a cardiopulmonary bypass was established, with femoral arterial cannulation for return and the left atrial appendage cannulation for drainage. The false aneurysm, surrounded by inflammatory tissue, was adherent to the left upper lobe and extended proximally just below the left subclavian artery take-off. The patient was thus cooled to 18°C so that hypothermic circulatory arrest could be performed. The drainage was satisfactory until the heart fibrillated, then due to some degree of aortic regurgitation, the left ventricle (LV) had a tendency to distend which required a further LV apical vent and a separate cannulation to the left-sided SVC with a right-angled cannula. On reaching 18°C, the left lung was carefully mobilized, however, significant bleeding from the false aneurysm occurred and therefore the circulation was stopped. Inspection of the false aneurysm showed that the patch was totally dehisced within, with a proximal neck just distal to the origin of the left subclavian artery identified. A 22 mm Gel seal tube graft was anastomosised end-to-end using continuous 3/0 proline, with a distal anastomosis constructed to follow, still being well within the safe limit period of hypothermic circulatory arrest. Having completed the anastomosis, de-airing maneuvers were performed and the circulation was restarted. The patient was re-warmed and the heart was defibrillated successfully at 29°C. On completion of re-warming, cardiopulmonary bypass was discontinued uneventfully on minimal isotropic support. Having secured hemostasis including the raw surface of the left lung, the chest was closed in layers with 2 drains.
The patient returned to the intensive care unit in stable hemodynamic condition on minimum inotropic support (dopamine 5 mcg·kg–1·min–1), and trasylol infusion. Extubation occurred at 12 hours with no neurological deficit. Chest drains were removed on 3rd postoperative day and the patient was discharged on day 5.
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DISCUSSION
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Aneurysm formation in the aortic isthmus after repair may be preconditioned by congenital (thinning or cystic medionecrosis) or acquired (atherosclerosis) changes of the aortic wall, as well as altered hemodynamics (hypertension or blood flow turbulence).1–2 Aneurysm can also be attributed to excessive excision of coarctation, damage to the intima, and increased aortic wall stress. 1–2 Hemodynamically, a rigid synthetic patch may cause dilatation of the more pliable aortic wall, whereas long standing hypertension causes additional stress and turbulence at the coarctation repair site.2 Other possible causes are excessive stress load on the suture line, particularly at the patch angles, suture line dehiscence, wear and fragmentation of the synthetic patch, infection, necrosis of the aorta due to an impaired blood supply from a dissection, or an excessive ligation.1–2
Although aneurysms are frequently asymptomatic, their clinical presentation may range from asymptomatic to sudden death due to sudden profuse bleeding.1–3 Possible presentations may also include sepsis or aorto-bronchial fistula leading to hemoptysis.1–4 Misleading clinical presentation may fatally delay the diagnosis, and thus highlights the importance of good history taking and the need of an earlier involvement of cardiac surgeons and cardiologists in the management of these patients.
There is a well-documented risk of aneurysm formation which is related to the use of prosthetic material and is probably unrelated to resection of the intimal ridge at the site of the stenosis.1–2 The incidence of late aneurysm formation after patch graft aortoplasty ranges from 27–89% and appears to be highest for repairs performed during the patient’s adult life.2 This highlights the importance of a proper follow-up in these patients, and indicates that professionals involved in the clinical care of patients who have undergone previous coarctation repair with patch graft aortoplasty should be aware of the late risk of false aneurysm occurrence. Suspicion of aneurysm development demands hospitalization, appropriate investigations and in the case of confirmed diagnosis, re-operation should be mandatory.
As the current case shows, the use of chest radiograph, echocardiogram and computed tomography are unreliable for diagnosis of this complication. This is in keeping with the data from Bromberg et al who reported a sensitivity of echocardiography and chest computed tomography for detecting an aneurysm of 71% and 66%, and a specificity of 76% and 85%, respectively.5 On the contrary, MRI appears to be useful for follow-up assessment of aneurysm and restenosis and is therefore recommended for periodic screening of patients to prevent potentially fatal complications.2–3 It should also be noted that another possible investigation is aortography, however, this was avoided in our patient due to the history of allergic reaction to intravenous contrast media.
The risk of spinal cord ischemic injury in patients with aneurysmal disease of the aorta is substantial, particularly when no distal perfusion techniques are used.6 Often, due to the complicated presentation of the false aneurysm, the use of clamps is not an option, as this might lead to severe intraoperative hemorrhage.6 In such cases, protection of the spinal cord can safely be achieved with hypothermic circulatory arrest.6 This technique contributed to several advantages in the management of our case as it facilitated adequate exposure of the structures involved, providing protection from intraoperative hemorrhage, thus avoiding placement of clamps on fragile tissue, and providing adequate protection of the brain, spinal cord and other organs.5 Recently, the use of percutaneous placement of intravascular stents in patients afflicted by descending thoracic aortic aneurysm disease is becoming an option. Karmy-Jones et al recently reported a case in which endovascular occlusion of a fistula between a thoracic aortic pseudoaneurysm and the lung was successfully managed by placement of an aortic endovascular stent-graft.4 This is obviously a promising technique and its application may well be widespread in the future.
In conclusion, aortic coarctation repair with a patch graft aortoplasty may be complicated by late occurrence of pseudoaneurysm. This complication may be fatal if diagnosis is delayed, and any professional involved in the clinical care of these patients should be aware of this risk. We strongly recommend systematic yearly follow-up with magnetic resonance imaging with input from a cardiologist/cardiac surgeon.
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REFERENCES
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- Bromberg BI, Beekman RH, Rocchini AP, Snider AR, Bank ER, Heidelberger K, et al. Aortic aneurysm after patch aortoplasty repair of coarctation: a prospective analysis of prevalence, screening tests and risks. J Am Coll Cardiol. 1989;14:734–41.[Abstract]
- Rokkas C, Murphy S, Kouchoukos N. Aortic coarctation in the adult: Management of complications and coexisting arterial abnormalities with Hypothermic cardiopulmonary bypass and circulatory arrest. J Thorac Cardiovasc Surg 2002;124:155–61[Abstract/Free Full Text]
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ABSTRACT
INTRODUCTION
