Abstract
Background
Infective aortitis is a rare complication following coarctoplasty. Unrecognized concomitant congenital defects such as patent ductus arteriosus (PDA) could be devastating.
Case Summary
We describe a young woman presenting with constitutional symptoms, elevated inflammatory markers, and a history of coarctoplasty with stent a decade earlier. Imaging revealed a large previously overlooked PDA and an aortic pseudoaneurysm distal to the stent covered by vegetations. Positron emission tomography/computed tomography demonstrated metabolically active lesions within the pulmonary artery and spleen. Broad-spectrum antibiotics, surgical stent excision, pseudoaneurysm repair with homograft, and PDA ligation achieved favorable outcomes with no residual infection on follow-up.
Discussion
Sophisticated diagnostic modalities and a multidisciplinary approach are essential in rare, life-threatening complications. With growing use of endovascular therapies for aortic coarctation, awareness of uncommon complications and their management is crucial.
Take-Home Messages
A comprehensive preprocedural assessment to detect coexisting anomalies is essential. Multimodality imaging is key for diagnosis, treatment planning, and follow-up. Combined antibiotic therapy and surgical intervention ensured complete recovery
Key words: congenital heart disease, coarctation of aorta, coarctoplasty, patent ductus arteriosus, aortitis
Visual Summary
History of Presentation
Infective aortitis is a serious and potentially fatal complication following coarctoplasty. We describe a case of an infected aortic stent at the site of prior coarctation repair.
Take-Home Messages
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Comprehensive preprocedural assessment to detect coexisting anomalies is essential.
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Multimodality imaging is key for diagnosis, treatment planning, and follow-up.
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Combined antibiotic therapy and surgical intervention ensured complete recovery.
A 29-year-old female with prolonged malaise, anorexia, and intermittent fever was referred to our center. She had undergone coarctoplasty with stent 10 years earlier. Her symptoms persisted despite multiple courses of oral antibiotics prescribed during several outpatient visits in the past 2 months.
On admission, she appeared ill but was hemodynamically stable. Cardiac auscultation revealed a continuous systolic murmur was heard at the upper left sternal border. Symmetrically diminished lower-extremity pulses were also noted. No cutaneous stigmata of endocarditis were present. Laboratory evaluation showed a leukocyte count of 5,500/μL with neutrophil predominance (69%), anemia (hemoglobin, 8.7 g/dL), and elevated erythrocyte sedimentation rate (93 mm/h) and C-reactive protein (38 mg/L).
Past Medical History
The patient had undergone stent coarctoplasty 10 years prior.
Differential Diagnosis
In a patient with prolonged fever, constitutional symptoms, elevated inflammatory markers, and a history of coarctoplasty with stent, the most leading differential diagnosis is infective aortitis secondary to stent infection or native valve endocarditis. Other considerations include large-vessel vasculitis (eg, Takayasu, Behçet's), disseminated tuberculosis, and less likely, noninfectious causes such as thromboembolism or intravascular lymphomas. The presence of a prosthetic vascular device and multiterritory arterial lesions favored an infectious etiology.
Investigations
Echocardiography revealed a large patent ductus arteriosus (PDA) with a left to right shunt (Figure 1A2, Video 1), as well as a sizable pseudoaneurysm surrounding the distal portion of the aortic stent (Figure 1A1, Videos 2 and 3). Multiple large, mobile echodensities were visualized extending from the PDA site into the left pulmonary artery (LPA) along with severe stent stenosis harboring several small, mobile echodensities on its surface (Figure 1A3, Videos 4 and 5).
Figure 1.
Echocardiography Images
(A1) X-plane transesophageal echocardiography view showing large pseudoaneurysm (∗) at the distal part of the aortic stent (arrow) in addition to multiple small vegetations attached to the stent. The stent vegetations can also be appreciated in the Videos 2 and 3. (A2) Parasternal short-axis transthoracic echocardiography view indicating patent ductus arteriosus (PDA) flow from the descending aorta into the left pulmonary artery (red arrow). The marked circular sign is presenting the coarctation stent in the descending aorta. (A3) Two-dimensional plain view echocardiography demonstrating 2 vegetations in the left pulmonary artery (∗) originating from PDA site. The red arrow is indicating the ascending aorta.
Abdominal ultrasonography showed massive splenomegaly (210 mm) with few hypoechoic lesions suggestive of infarctions. Computed tomography angiography of the aorta confirmed a pseudoaneurysm of the descending aorta distal to the stent (Figure 2B1 to 2B). Additional pseudoaneurysms involving the splenic and left internal iliac arteries were also identified (Figure 3C1 to 3C).
Figure 2.
Thoracic Computed Tomography Angiography Images
(B1) CT angiography shows a large filling defect (22∗8 mm) in the left pulmonary artery (red arrow) corresponding to vegetation/thrombosis based on echocardiographic findings. (B2) Aortic CTA: CT angiography demonstrating a large pseudoaneurysm (✴) (46∗32∗49 mm) at the descending aorta at the distal part of the aortic stent (open arrow) (patent ductus arteriosus [PDA] marked with arrow).
Figure 3.
Abdominal CT Angiography
(C1 and C2) CT angiography images demonstrating mycotic aneurysms at the splenic artery (11∗8 mm) and left internal iliac artery (23∗15 mm), respectively. (C3) CT angiography image demonstrating an enlarged spleen with mass-like lesions in favor of infarctions or abscess formation.
The 18F-fluorodeoxyglucose positron emission tomography (PET) scan was consequently performed to determine the extent of systemic involvement which revealed increased metabolic activity in the LPA, indicative of an ongoing infectious process, along with pulmonary opacities in the right lung, possibly indicative of septic emboli (Figures 4A and 4B). Splenomegaly with multiple rim-enhancing hypermetabolic lesions was also identified (Figure 5).
Figure 4.
Positron Emission Tomography Image of the Lung
(A) 18F-FDG PET/CT scan: Increased FDG uptake in left pulmonary artery (green arrow) corresponding to vegetation/thrombosis (left column) and 6-month postsurgery follow-up (right column). (B) 18F-FDG PET/CT reveal a couple of FDG-avid pulmonary opacities in right lung (green arrows) suspicious for active infectious process possibly secondary to septic emboli; and with 6-month postsurgery follow-up (right column).
Figure 5.
Positron Emission Tomography Image of the Spleen
18F-FDG PET/CT scan: Overall regression in size and FDG uptake over time (ie, 6-month follow-up) (left to right), more likely secondary to splenic infarction along with possible infected areas.
Management (Medical/Interventions)
Given the high suspicion for aortic stent infection, empiric antibiotic therapy comprising imipenem, rifampin, and vancomycin was initiated in accordance with the experts' opinion and available recommendation.1,2 Blood cultures (both short and long incubation periods) and polymerase chain reaction assays for brucellosis and Mycobacterium tuberculosis were negative.
After a 4-week course of antibiotics, the patient showed marked clinical improvement, and significant reduction in metabolic activity was observed.
She subsequently underwent surgical intervention through a left thoracotomy approach. The infected stent and pseudoaneurysm were excised, followed by pulmonary artery thrombus removal and PDA ligation. Reconstruction was accomplished using homograft conduits anastomosed to the aortic arch and descending aorta (Supplemental Figure S3-4). The patient had an uneventful postoperative course.
Outcome and Follow-Up
The surgically excised aortic stent's culture was negative, but polymerase chain reaction confirmed positive for “Stenotrophomonas maltophilia,” and targeted antibiotic therapy was subsequently tailored accordingly.
Hypermetabolic lesions of spleen regressed in size and fluorodeoxyglucose uptake over the 3-month follow-up period (Figure 2C). She remained asymptomatic during the 1-year follow-up with no sign of infection in serial imaging studies (Figure 6).
Figure 6.
Computed Tomography Angiography
(A) The infected stent at the coarctation site. Note the overlooked patent ductus arteriosus (PDA) marked with a red arrow. (B) CT angiography of the aorta 1 year after surgery. Pseudoaneurysm was excised, PDA was ligated, and interposition aortic homograft between the distal arch and descending thoracic aorta was seen with no signs of infection or complication.
Discussion
Transcatheter coarctoplasty with a stent has emerged as a promising alternative to surgical repair, offering excellent short- and long-term outcomes.2,3 Although rare, stent infection represents one of the most serious complications, often necessitating open surgical repair.4 Metallic scaffolds can serve as a nidus promoting bacterial adhesion, and coexisting congenital vascular anomalies may create a permissive environment for the onset of aortitis.4,5 Mortality associated with infectious aortitis ranges from 21% to 44%.4
Echocardiography remains the primary imaging modality to diagnose infective endocarditis.5 Magnetic resonance angiography and computed tomography angiography provide complementary anatomical detail.2 PET/computed tomography scan serves as an ideal supplementary method for suggesting stent infection,4 whereas a heterogeneous and intense uptake pattern in contrast to a diffuse and homogeneous one will raise concern for infection.6 In addition, serial PET scans can assist clinicians for accurate monitoring of treatment efficacy over time.6
Management of stent infection and related aortitis remains still challenging due to the absence of robust high-quality evidence. Nonetheless, surgical repair with the removal of infected zones combined by broad-spectrum antibiotic therapy remains the best therapeutic solution.
Conclusions
This case underscores the importance of a comprehensive preprocedural evaluation before coarctoplasty, particularly to identify coexisting congenital anomalies. The overlooked PDA likely contributed to persistent abnormal blood flow at the stent site, promoting endothelial injury and facilitating bacterial seeding. In the presence of a metallic scaffold, these changes provided an ideal nidus for microbial adherence and biofilm formation following transient or sustained bacteremia. The case also emphasizes the pivotal role of multimodality imaging in both diagnosis and treatment planning. Prompt initiation of broad-spectrum antibiotics followed by surgical repair and careful postoperative surveillance is essential for a successful management.
Funding Support and Author Disclosures
The authors have reported that they have no relationships relevant to the contents of this paper to disclose.
Footnotes
The authors attest they are in compliance with human studies committees and animal welfare regulations of the authors’ institutions and Food and Drug Administration guidelines, including patient consent where appropriate. For more information, visit the Author Center.
Appendix
Visual Summary.
| Timeline | Intervention | Events |
|---|---|---|
| Day 1 | Admission, initial assessment | 29-year-old woman admitted with prolonged malaise, anorexia, and fever. She had a history of coarctoplasty with stent 10 years ago. She presented with stable hemodynamics, continuous systolic murmur at the ULSB. Splenomegaly was noted. |
| Day 1, 2 | Laboratory testing | Baseline laboratory testing demonstrated anemia (Hgb, 8.7 g/dL) and increased inflammatory markers (ESR, 93 mm/h; CRP, 38 mg/L) Blood cultures were negative for aerobic and anaerobic germs. Urine culture was negative. PCR for Covid-19, Entrococci, non-Mycobacterium tuberculosis, Strep pneumonia, Staphylococcus aureus, Brucella, tuberculosis, fungi were negative. |
| Day 1 | Imaging, diagnosis | Echocardiography revealed PDA, large pseudoaneurysm around the distal part of the stent, and vegetations. |
| Day 1 | Antibiotic therapy, treatment | Broad empiric spectrum antibiotic therapy comprising of imipenem, rifampin, and vancomycin were started. |
| Day 2 | Imaging, diagnosis | Abdominal ultrasonography showed a large spleen with infarction. |
| Day 3 | Imaging, diagnosis | CTA of aorta demonstrated pseudoaneurysm of the descending aorta at the distal part of the stent, and pseudoaneurysm of splenic and left internal iliac arteries. |
| Day 3 | Imaging, diagnosis | PET scan indicates a hypermetabolic activity in LPA, septic emboli in the right lung, and hypermetabolic lesions in the spleen. |
| Day 42 | Surgery, treatment | Pseudoaneurysm and stent excision, PDA ligation, aortic homograft anastomosis was performed. |
| Day 43 | Laboratory testing | Excised-stent PCR was positive for Stenotrophomonas maltophilia |
| Day 43 | Antibiotic therapy, treatment | Proper antibiotic therapy according to the positive PCR result was initiated. |
| Day 62 | Imaging, follow-up | PET/CT scan showed reduction in the size of hypermetabolic lesions in the lung and spleen. |
Equipment List.
| Imaging |
X8-2t xMATRIX transesophageal transducer
S4-2 Phased Array transducer
|
| Surgery | Patent ductus arteriosus closure Infected stent and aortic pseudoaneurysm excision Descending aorta replacement with 2 aortic homograft |
Contributor Information
Saeid Hosseini, Email: saeid.hosseini@yahoo.com.
Melody Farrashi, Email: m.farrashi@gmail.com.
Appendix
Transthoracic Echocardiography Parasternal Short-Axis View Demonstrating Patent Ductus Arteriosus Flow
X-Plane Transesophageal Echocardiography Showing Large Pseudoaneurysm at the Distal Part of the Aortic Stent With Multiple Small Mobile Vegetations Attached to the Stent
Three-Dimensional Transesophageal Echocardiography Demonstrating Large Pseudoaneurysm and Multiple Vegetations on the Infected Stent
Transthoracic Echocardiography Parasternal Short-Axis View Showing Large Vegetations in Left Pulmonary Artery Originating From Patent Ductus Arteriosus
X-Plane Transesophageal Echocardiography View Demonstrating Large Vegetations in Left Pulmonary Artery Originating From Patent Ductus Arteriosus
Echocardiographic Evidence of Stent Stenosis Supplemental Figure 3 and 4: Images of Operative View and the Excised Stent.
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Associated Data
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Supplementary Materials
Transthoracic Echocardiography Parasternal Short-Axis View Demonstrating Patent Ductus Arteriosus Flow
X-Plane Transesophageal Echocardiography Showing Large Pseudoaneurysm at the Distal Part of the Aortic Stent With Multiple Small Mobile Vegetations Attached to the Stent
Three-Dimensional Transesophageal Echocardiography Demonstrating Large Pseudoaneurysm and Multiple Vegetations on the Infected Stent
Transthoracic Echocardiography Parasternal Short-Axis View Showing Large Vegetations in Left Pulmonary Artery Originating From Patent Ductus Arteriosus
X-Plane Transesophageal Echocardiography View Demonstrating Large Vegetations in Left Pulmonary Artery Originating From Patent Ductus Arteriosus
Echocardiographic Evidence of Stent Stenosis Supplemental Figure 3 and 4: Images of Operative View and the Excised Stent.