Recurrent aortic aneurysms following thoracic aortic stent-graft repair in a patient with Cogan syndrome.
OBJECTIVE
To report the need for multiple surgical interventions to treat recurrent aortic aneurysms in a patient with Cogan syndrome.


METHODS
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.


CONCLUSIONS
This case highlights the pitfalls of stent-graft repair in a patient with presumed connective tissue disease.
Su
Jang Wen
JW
Department of Cardiothoracic Surgery, National Heart Centre, 17 Third Hospital Avenue, Singapore 168752. bottle1001@yahoo.com
Low
Andrea Hsiu Ling
AH
Tay
Kiang Hiong
KH
Sebastian
Matthew G
MG
Thumboo
Julian
J
Sin
Kenny Yoong Kong
KY
D017541:Aneurysm, False / Q000175:diagnosis / Q000209:etiology / Q000628:therapy; D000800:Angioplasty, Balloon / Q000379:methods*; D017545:Aortic Aneurysm, Thoracic / Q000175:diagnosis / Q000209:etiology / Q000628:therapy; D001025:Aortitis / Q000150:complications; D001027:Aortography; D019917:Blood Vessel Prosthesis Implantation / Q000379:methods*; D006311:Hearing Disorders / Q000150:complications; D018611:Heart Bypass, Left; D006801:Humans; D007634:Keratitis / Q000150:complications; D008279:Magnetic Resonance Imaging; D008297:Male; D012008:Recurrence; D012086:Reoperation; D015607:Stents; D013577:Syndrome; D013908:Thoracotomy; D014057:Tomography, X-Ray Computed; D014717:Vertigo; D015837:Vestibular Diseases / Q000150:complications