Ann Thorac Surg 2006;81:339-341
© 2006 The Society of Thoracic Surgeons
Case report
Infectious Ductal Aneurysm After Coil Embolization in an Infant
Takeshi Shinkawa, MD
*
,
Masaaki Yamagishi, MD,
Keisuke Shuntoh, MD,
Katsuji Fujiwara, MD,
Taiji Watanabe, MD,
Satomi Yoshida, MD
Department of Pediatric Cardiovascular Surgery, Children's Research Hospital, Kyoto Prefectural University of Medicine, Kyoto, Japan
Accepted for publication September 24, 2004.
* Address correspondence to Dr Shinkawa, Department of Pediatric Cardiovascular Surgery, Children's Research Hospital, Kyoto Prefectural University of Medicine, Kawaramachi, Hirokoji, Kamigyo-ku, Kyoto 6028566, Japan (Email: newriver{at}koto.kpu-m.ac.jp).
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Abstract
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We report a case of ductal aneurysm after transcatheter coil embolization in an infant. The aneurysm was asymptomatic and was not detected until it ruptured during surgical intervention. We suspect that the aneurysm was induced by methicillin-resistant Staphylococcus aureus infection, as the resected aneurysmal wall was severely infiltrated by inflammatory cells and the patient had recurrent methicillin-resistant Staphylococcus aureus infection. We recommend remaining on guard against formation of a ductal aneurysm after coil embolization, especially in patients with associated recurrent bacteremia.
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Introduction
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Transcatheter coil embolization of the patent ductus arteriosus is widely used to treat patients with patent ductus arteriosus and pulmonary hypertension. We describe a 9-month-old girl who developed a ductal aneurysm after coil embolization.
One week after birth, a girl with situs inversus, double-outlet right ventricle, large remote type ventricular septal defect, transposition of the great arteries, infundibular pulmonary stenosis, patent ductus arteriosus, and pulmonary hypertension developed necrotizing enterocolitis. She was treated with imipenem with immediate improvement in her inflammation, but a barium enema examination performed at 1 month of age showed severe stenosis of the sigmoid colon. At the age of 2 months, she was referred to our hospital. She had septic shock caused by methicillin-resistant Staphylococcus aureus (MRSA) and was treated with vancomycin and exchange transfusions. At the age of 3 months, she underwent resection of the stenotic colon and creation of a stoma. Frequent recurring respiratory infections and uncontrollable heart failure persisted, leading to a cardiac catheter examination, which was performed at 5 months of age and which revealed pulmonary hypertension with high pulmonary blood flow. To decrease pulmonary blood flow, we embolized the patent ductus arteriosus using a detachable coil with three 5-mm loops (Flipper, Cook, IN) at the age of 7 months (Fig 1). The patient's general condition did not improve because of persistent pulmonary hypertension, and she had occasional hypoxic spells due to dynamic infundibular pulmonary stenosis. At 8 months of age, she again developed MRSA sepsis, which was treated as before.

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Fig 1. Lateral aortogram showing occlusion of the patent ductus arteriosus after placement of a screwing detachable coil. No ductal aneurysm is present.
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At 9 months of age, an echocardiogram showed no intracardiac vegetation or evidence of a ductal aneurysm. At this time, at a weight of 4.2 kg, we performed surgery to stabilize pulmonary blood flow by ligating the main pulmonary artery and placing a Blalock-Taussig shunt. At the time of median sternotomy, cloudy pericardial fluid was cultured and later grew MRSA. While dissecting the great vessels, massive bleeding from the left side of the aorta occurred, and we first noticed a giant ductal aneurysm that had ruptured (Fig 2, A). Cardiopulmonary bypass was established and the aneurysm was resected (Fig 2, B). The Flipper coil was at the pulmonary stump of the aneurysm, but no vegetation was found in this tissue (Fig 3). A right modified Blalock-Taussig shunt was placed and the main pulmonary artery was ligated. Weaning from bypass was uneventful, and the patient's postoperative course was satisfactory without evidence of recurrent infection. She is currently waiting for a Fontan operation.

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Fig 2. Intraoperative views. (A) Large ductal aneurysm (5 x 4 cm) with a very thin wall that ruptured during dissection (white arrows). (B) Ductal aneurysm having a fusiform and normal-looking neck on the aortic side (black arrow).
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Fig 3. Resected ductus showing the aneurysmal formation with a diameter of 30 mm and the detachable coil at the pulmonary stump (black arrow). Microscopic findings of the aneurysmal wall showed severe destruction of elastic fibers in the media and severe inflammatory cell infiltration and formation of granulation tissue.
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Microscopic examination of the resected aneurysmal wall showed severe destruction of elastic fibers in the media and severe inflammatory cell infiltration with formation of granulation tissue; however, no microorganisms such as cocci or fungi were seen or grew on culture.
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Comment
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Percutaneous coil embolization to close the patent ductus arteriosus is widely accepted as a therapeutic procedure for symptomatic patients with patent ductus arteriosus and pulmonary hypertension. This procedure is considered safer and less invasive than surgical intervention, and reports of severe complications are rare [1, 2]. We here report a postcoil embolization ductal aneurysm caused by infection. To date, the only other reported case of a postcoil embolization ductal aneurysm was in a patient with Marfan syndrome without infection [3].
In the present case, the ductal aneurysm rapidly developed in the 2 months between coil embolization and surgical intervention. The aneurysm presumably resulted from weakening and thinning of the ductal wall as a consequence of destruction of the wall's elastic fibers with infiltration of inflammatory cells and exposure of the aortic part of the ductus to high aortic pressure. Although no pathogen was detected in the resected aneurysmal wall and no microorganisms could be grown from the resected aneurysmal wall, we suspect an MRSA infection because of the presence of this organism in the pericardial effusion. The presence of granulation tissue, heavy infiltration of inflammatory cells, and the fragility of the aneurysmal tissue favored the diagnosis of an infectious ductal aneurysm. We had difficulty controlling the MRSA infection before performing the coil embolization but staphylococcal sepsis recurred after the procedure. The coil and the resulting turbulent blood stream probably made conditions in the ductus arteriosus favorable for the blood-borne MRSA, considering that infectious endocarditis and blood-borne Staphylococcus organisms can cause pulmonary mycotic aneurysm, especially in patients with congenital heart disease [4, 5]. The presence of a foreign body such as a detachable coil probably increases the infection risk.
In conclusion, this is the first report of a postcoil embolization ductal aneurysm caused by an infection. We recommend remaining on guard against formation of a ductal aneurysm after coil embolization, especially in patients with associated recurrent bacteremia.
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References
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- O'Donnell C, Neutze JM, Skinner JR, Wilson NJ. Transcatheter patent ductus arteriosus occlusionevolution of techniques and results from the 1990s. J Paediatr Child Health 2001;37:451-455.[Medline]
- Prieto LR, DeCamillo DM, Konrad DJ, Scalet-Longworth L, Latson LA. Comparison of cost and clinical outcome between transcatheter coil occlusion and surgical closure of isolated patent ductus arteriosus Pediatrics 1998;101:1020-1024.[Abstract/Free Full Text]
- Marasini M, Rimini A, Zannini L, Pongiglione G. Giant aneurysm following coil occlusion of patent ductus arteriosus Catheter Cardiovasc Interv 2000;50:186-189.[Medline]
- Bozkurt AK, Oztunc F, Akman C, Kurugoglu S, Eroglu AG. Multiple pulmonary artery aneurysms due to infective endocarditis Ann Thorac Surg 2003;75:593-596.[Abstract/Free Full Text]
- Caplin JL, Dymond DS, Barrett DS, Rees RS, Rees GM, Spurrell RA. Pulmonary mycotic aneurysms secondary to infective endocarditis in a patient with a persistent ductus arteriosus and partial anomalous pulmonary venous drainage Eur Heart J 1985;6:985-988.[Abstract/Free Full Text]
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