Pediatric Cardiology Fellow Children's Hospital of Michigan, United States
Abstract:
Background: Mycotic aneurysm (MA) has been reported mostly in adults after repair of aortic arch dissection. However, development of bacterial MA following cardiac surgical repair is rarely seen in children. We report the first case of development of fungal MA in an infant following the surgical repair of anomalous origin of the left coronary artery arising from the pulmonary artery (ALCAPA).
Case: 2 months old female presented to our emergency department with respiratory distress and was found to have ALCAPA for which she underwent repair by direct transfer of the anomalous coronary artery. Post-operative course was remarkable for Serratia respiratory infection and bacteremia. On postoperative day 32, a pre discharge echocardiogram showed a large aneurysm measuring at least 3cm x 3cm arising from the ascending aorta and anterior to the right ventricle (Figure1). A computed tomography angiogram (CT scan) confirmed the ascending thoracic aorta aneurysm, extending anteriorly measuring 3.5 x 4.2 x 3.1 cm (Figure 2). She was taken urgently to the operating room and had resection and repair of the large aortic pseudoaneurysm with pulmonary homograft. Mucoid-like tissue at the junction of the thick wall of the aneurysm and the suture line of the ascending aorta was noted. Tissue of the aneurysm wall sent for bacterial and fungal cultures as well as repeat blood cultures were negative. She was empirically treated with IV antibiotic for 4 weeks. Four weeks after surgery, echocardiogram showed recurrent large ascending aortic arch pseudoaneurysm measuring 3.5 x 3.9 x4.0 cm confirmed by CT scan. She again underwent repair of ascending aortic arch pseudoaneurysm with aortic homograft and reimplantation of the left coronary artery. Aortic wall tissue sent for pathology showed fungal hyphae (Figure 3). Fungal PCR was positive for Rhizopus microspores. She was treated with IV liposomal amphotericin for 3 months followed by oral posaconazole for 3 more months. Extensive immunodeficiency work up was negative and the source of the fungus could not be found. She was transitioned to oral Posaconazole for additional three months as outpatient. Repeat chest CT scan one month following antifungal treatment showed no recurrent aortic pseudoaneurysm. The patient is currently clinically stable.
Conclusion: Multidisciplinary collaboration, aggressive surgical interventions and long-term antifungal therapy resulted in favorable outcome in our first case reported of mycotic aneurysm of the aorta caused by Rhizopus micropores following cardiac repair.
