Recurrent aortic aneurysms following thoracic aortic stent-graft repair in a patient with Cogan syndrome.

Abstract

PURPOSE:

To report the need for multiple surgical interventions to treat recurrent aortic aneurysms in a patient with Cogan syndrome.

CASE REPORT:

A 17-year-old Chinese man with clinical Marfanoid features had a left common carotid artery pseudoaneurysm electively repaired with an autologous saphenous vein graft. Four months later, he presented with acute chest pain. Computed tomography (CT) revealed a 1-cm pseudoaneurysm at the mid descending aorta; a 24 x 100-mm Talent stent-graft was implanted to exclude the pseudoaneurysm. He was also found to have increasing left-sided hearing loss. A month later, the patient was re-admitted with vertigo and keratitis, which were treated appropriately. Nine months following stent-graft insertion, he was admitted with acute hemoptysis. Urgent CT showed a rupture at the proximal end of the stent-graft, with hemorrhage into the lung parenchyma. In an emergent procedure, the stent-graft was removed, and the descending thoracic aorta was repaired. Intraoperatively, a large pseudoaneurysm was found arising from the proximal part of the stented aorta, which appeared thickened. His postoperative recovery was uneventful. Nine months after the thoracotomy, a routine CT revealed an aneurysm at the distal descending thoracic aorta. On re-thoracotomy, a de novo saccular aneurysm was found 2.5 cm from the distal anastomosis. The affected segment was replaced with a Dacron graft. The distal aorta appeared thickened and edematous; histology confirmed aortitis. The patient was subsequently diagnosed with Cogan syndrome and given corticosteroids and methotrexate. There is no evidence of recurrence at nearly 2 years after the last intervention.

CONCLUSION:

This case highlights the pitfalls of stent-graft repair in a patient with presumed connective tissue disease.

• Authors: Jang Wen Su, Andrea Hsiu Ling Low, Kiang Hiong Tay, Matthew G Sebastian, Julian Thumboo, Kenny Yoong Kong Sin
• Journal: Journal of endovascular therapy : an official journal of the International Society of Endovascular Specialists (J Endovasc Ther) Vol. 13 Issue 6 Pg. 779-82 (Dec 2006) ISSN: 1526-6028 [Print] United States
• PMID: 17154705 (Publication Type: Journal Article)

Topics

• Aneurysm, False (diagnosis, etiology, therapy)
• Angioplasty, Balloon (methods)
• Aortic Aneurysm, Thoracic (diagnosis, etiology, therapy)
• Aortitis (complications)
• Aortography
• Blood Vessel Prosthesis Implantation (methods)
• Hearing Disorders (complications)
• Heart Bypass, Left
• Humans
• Keratitis (complications)
• Magnetic Resonance Imaging
• Male
• Recurrence
• Reoperation
• Stents
• Syndrome
• Thoracotomy
• Tomography, X-Ray Computed
• Vertigo
• Vestibular Diseases (complications)