Relapse of ascending aortic pseudoaneurysm and aortopulmonary fistula after first resection: a case description

Introduction

Ascending aortic pseudoaneurysm (AAP) is an uncommon condition, affecting fewer than 0.5% of patients undergoing cardiothoracic surgery (1). In this article, we report an unusual case of a patient who developed an AAP with a pulmonary fistula after the first surgical treatment. This article seeks to provide comprehensive insights into the various causes and surgical methods for managing AAP, emphasizing the potential risk and seriousness of such conditions developing in younger individuals.

Case presentation

A 14-year-old male was admitted to the Qindao University Medical College Affiliated Yantai Yuhuangding Hospital for chest pain, tightness, and breath-holding. A continuous ejection murmur was noted in the physical examination. Transthoracic echocardiography revealed an abnormal vascular communication measuring 4.0 mm (Figure 1A) connecting AAP (Figure 1B) with the pulmonary artery, along with a continuous turbulent high-velocity flow (both systolic and diastolic) between the pulmonary artery and AAP. Computed tomography angiography (CTA) revealed an irregularly shaped mixed-density mass measuring 4.1 cm by 4.5 cm, indicative of a pseudoaneurysm, and suggestive of a fistulous trajectory between the aorta and pulmonary artery (Figure 2).

Figure 1 Preoperative ultrasonic cardiogram. (A) The arrow points to the aortopulmonary fistula; (B) the arrow points to the ascending aortic pseudoaneurysm.

Figure 2 Preoperative enhanced computed tomography of the chest; the arrow points to the ascending aortic pseudoaneurysm.

There was no evidence of any obvious infection. The patient underwent bacterial culture and rheumatoid immune index tests, and mycobacterium tuberculosis test, showing no positive results. The multidisciplinary team decided on open surgery (2) based on the anatomical features, clinical presentation, and patient’s comorbidities. The patient underwent aortic aneurysm resection, ascending aorta angioplasty, ascending supra-aortic septal resection, pulmonary artery repair, and pulmonary valve exploration, and the aortic tissue was sent for pathological examination (Figure 3A,3B). The examination revealed an elliptical lesion with circumferential ulceration and raised inflammatory borders, demonstrating internal fibrous septations in the defect.

Figure 3 Pathological results (hematoxylin and eosin staining, ×40). (A) Cyst wall pathology; (B) pathology of cystic wall thrombus.

Six months later, the patient was admitted to hospital for the second time with chest tightness, and the postoperative echocardiogram suggested pseudoaneurysm resection, post-ascending aorta angioplasty (Figure 4A), AAP formation with wall attachment thrombus. The submitted specimen of the pseudoaneurysm wall displayed histologic features of proliferative fibrous capsular tissue formation combined with mixed thrombus components. CTA of the abdominal aorta showed post-angioplasty of the ascending aorta, ascending aortic aneurysm with arterial-aortic fistula formation, and thrombosis of the wall of the aneurysm (Figure 4B,4C). After multidisciplinary consultation, the patient’s etiology of the condition remained undetermined. Two days after admission, the patient’s blood pressure and heart rate progressively decrease, and the patient ultimately died. The causes of death were related to the rupture of the AAP and obstructive shock.

Figure 4 Postoperative examination. (A) Ultrasonic cardiogram; (B) enhanced computed tomography of the chest; the arrow points to the ascending aortic pseudoaneurysm; (C) chest enhanced computed tomography three-dimensional reconstruction; the arrow points to the ascending aortic pseudoaneurysm.

All procedures performed in this study were in accordance with the Helsinki Declaration and its subsequent amendments. The study was approved by Committee of Qindao University Medical College Affiliated Yantai Yuhuangding Hospital (No. 2024-562). Written informed consent was obtained from the patient’s legal guardians for publication of this article and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

Although there may be various causes, prior cardiac surgery, infections, and intrinsic tissue frailty appear to be the frequent etiology. Atik et al. (3) found that up to one third of all patients who developed a pseudoaneurysm of the thoracic aorta had endocarditis. In the present case, the patient was only 14 years old, and had no history of car accident, surgery, trauma, infection, or Marfan syndrome. The patient’s bacterial culture before surgery was also negative for mycobacterium tuberculosis and other bacteria.

This case report sought to explore the underlying etiology of AAP. Ferrari et al. (4) discovered that a pseudoaneurysm following surgery of the ascending aorta might arise from the dehiscence at the suture line, either at the proximal or distal anastomosis connecting the graft to the ascending aorta, or from the sutures located in the aortic annulus. Additionally, other factors contributing to the development of a pseudoaneurysm include graft infection, mediastinitis, a dissected native aorta, or tissue necrosis caused by biologic glue, which could potentially lead to recurrence. Except in cases with an active infection, the direct closure of the dehiscence between the aorta and the vascular graft could help to avoid this problem.

Traditionally, the primary approach for managing pseudoaneurysms has been surgical intervention; however, for patients considered to be at excessive surgical risk, the endovascular treatment of AAPs is becoming a promising alternative (5). In the present case, the patient was released from the hospital following surgery but experienced a recurrence of an AAP 6 months after the initial resection. Treatment should be considered a viable alternative to surgery for individuals at prohibitive risk, even in emergency situations. Gottardi et al. reported on the successful treatment of a large pseudoaneurysm arising from the ascending aorta using a readily available stent graft (6). Becker et al. presented a series of cases involving endovascular repair of anastomotic aneurysms and pseudoaneurysms in patients who underwent open repair of the ascending aorta and aortic arch (7). In situations that are both urgent and emergent, it is necessary to anticipate associated illnesses and fatalities; if surgical interventions are not pursued, these alternative therapies may be appropriate. Afzal et al. (8) reported a case in which a previously healthy 13-year-old boy diagnosed with traumatic pseudoaneurysm without indication of aortopulmonary fistula underwent endovascular aortic stenting. Follow-up echo showed the stents to be in a good position and confirmed its patency. It also affirmed the short-term safety and efficacy of thoracic endovascular aortic repair (TEVAR) in pediatric traumatic thoracic pseudoaneurysm patients; however, stent occlusion, pseudo-coarctation, and stent durability should also be considered when assessing whether the patient should undergo open surgery or TEVAR.

Conclusions

This case provides insights into the possibility and severity of AAP in young patients. Ultimately, changes in the treatment approach may lead to decreased rates of mortality during the perioperative period.

Acknowledgments

None.

Footnote

Funding: This study was supported by the Youth Foundation of Yantai Yuhuangding Hospital (No. 202317 to H.W.).

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://qims.amegroups.com/article/view/10.21037/qims-24-2277/coif). H.W. received funding from the Youth Foundation of Yantai Yuhuangding Hospital (No. 202317). The other authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the Helsinki Declaration and its subsequent amendments. The study was approved by Committee of Qindao University Medical College Affiliated Yantai Yuhuangding Hospital (No. 2024-562). Written informed consent was obtained from the patient’s legal guardians for publication of this article and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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