Calcified-Ductal-Aneurysm-with Severe
Aortic Regurgitation
Saket Agarwal, Sunil K Kaushal, Parvathi U Iyer,
Poonam Khurana,
Krishna S Iyer
Departments of Pediatric Surgery and Radiodiagnosis,
Escorts
Heart Institute and Research Centre, New Delhi
A rare case of calcified aneurysm of the ductus arteriosus with severe aortic regurgitation is presented. We believe this is the first report of such a case in the English literature.
(Indian Heart J 2005; 57: 172–174)Key Words:
Aortic regurgitation, Ductal aneurysm, Congenital heart diseaseD
uctus arteriosus aneurysm is a rare condition that can be associated with serious complications including thromboembolism, rupture and death. To the best of our knowledge the combination of severe aortic regurgitation (AR) and patent ductus arteriosus (PDA) aneurysm has not been reported. We report such a case in an adult who was successfully treated at our institution.Case Report
A 27-year-old man presented with history of palpitations for 20 years, and dyspnea on exertion (New York Heart Association class II) of 2 years duration. He had no history of chest pain, fatigue or syncope.
On examination he was well-built, with no pallor, cyanosis or pedal edema. The jugular venous pressure was within normal limits. Blood pressure was 130/40 mmHg supine. An early diastolic decrescendo murmur was audible in the left parasternal area.
A plain chest X-ray revealed an enlarged heart, prominent aorta and dilated main pulmonary artery (PA) with increased hilar vascularity. A calcified shadow was observed in the region of the ductus (Fig. 1). A transthoracic echocardiogram (TTE) showed a dilated and hypertrophied left ventricle (LV) with severe AR. Mobile vegetations were seen on the aortic valve. There was a PDA with a large aneurysm at its aortic end. The branch pulmonary arteries were normal. The estimated PA systolic pressure was 40 mmHg.
A multi-slice computerized tomographic (CT) angiogram revealed a large patent ductus with a calcified aneurysm measuring 5×4.3×3.6 cm causing splaying and compression of the right and left pulmonary arteries. No thrombus was seen in the aneurysm (Fig. 2).
Cardiac catheterization and angiography was deferred in view of mobile vegetations on the aortic valve and the clarity of the anatomy obtained on CT angiogram. One stage repair of both lesions through mid-sternotomy was planned.
Operative technique: At surgery, the findings included a mildly dilated aortic root and a tense dilated PA which was pushed anteriorly by the calcified duct aneurysm. The entire wall of the ductus was calcified except for a rim of 5 mm at the PA end. The branch pulmonary arteries were normal in caliber, but stretched across the aneurysm. The ductus was dissected and looped around the pulmonary end. The patient was placed on cardiopulmonary bypass with ascending aortic and two-stage venous cannulation. The ductus was then carefully snugged and core cooling to 18°C was commenced. At 32°C the aorta was clamped, a transverse aortotomy was done and coronary ostial cardioplegia administered. The aortic valve was tricuspid and there were healed mobile vegetations on its right and non-coronary cusps. Aortic valve replacement was done using 21M St Jude Regent valve. The aortotomy was closed and root cardioplegia administered as core temperature reached 18°C. Circulatory arrest was established. The ductus was then transsected at the pulmonary end and the pulmonary artery bifurcation was dissected free from the anterior wall of the aneurysm. The aneurysm was then split open anteriorly to expose the aortic opening from within. Calcium-free margins of the aortic end were identified and these were closed with an oval piece of Meadox vascular graft (Boston Scientific, Meadox Medicals Inc., Oakland, NJ) measuring 6×4 cm using running 3/0 prolene suture. Before tightening the suture line, blood was returned to the patient through the arterial line and the aorta was de-aired. Cardiopulmonary bypass was resumed and re-warming started. The pulmonary artery was reconstructed using a pericardial patch, while re-warming was completed. The patient was successfully weaned off bypass on minimal inotropic support. Cardiopulmonary bypass time was 153 min, aortic cross clamp time was 76 min and circulatory arrest time was 25 min.
A post-operative CT angiogram revealed satisfactory exclusion of the aneurysm (Fig. 3).
Discussion
Surgical experience with aneurysm of the ductus arteriosus in adult is scant. Yoshitaka et al.
1 reported the case of a giant aneurysm of the ductus arteriosus with severe mitral regurgitation in a 58-year-old adult, which was repaired in two stages utilizing circulatory arrest.Aneurysm of the diverticulum of the ductus has been reported more frequently, with surgical closure being relatively more straightforward as only the narrow mouth of the diverticulum has to be tackled. Mitchell et al.2 reported successful management of 5 cases of aneurysm of diverticulum of the ductus in adult patients.
A calcified aneurysm of the ductus is a difficult entity to manage. The risks of thromboembolism and rupture are real although rare, and merit early intervention. The challenge in this case was that both lesions had to be managed simultaneously. The AR was significant enough to preclude prior management of the ductus aneurysm through a thoracotomy. The mid-sternotomy approach necessitated control of duct flow upon initiation of cardiopulmonary bypass. We were lucky to find a small area of non-calcified ductus at the pulmonary artery end which could be occluded at the onset of cardiopulmonary bypass.
The pre-operative CT angiogram clearly defined the lesion and helped us formulate a plan for management which was efficiently executed on the operating table. It defined the areas which were free of calcium, enabling us to get control of the ductus, and suture the patch. We believe this modality has certainly enhanced the accuracy of diagnosis and should play an increasing role in the management of such difficult patients in the future.
Correspondence:
References
Yoshitaka H, Hata T, Tsushima Y, Matsumoto M, Hamanaka S, Chikazawa G, et al. A case report of aneurysm of the ductus arteriosus combined with mitral regurgitation.
Mitchell RS, Seifert FC, Miller DC, Jamieson SW, Shumway NE. Aneurysm of the diverticulum of the ductus arteriosus in the adult. Successful surgical treatment in five patients and review of the literature.