Authors
Jansen, MichaelSaleh, Sheikh
Bolster, Margot
O'Donnell, Aonghus
Ahern, Thomas
Spence, Liam
Sheppard, Mary N
Burke, Louise
Affiliation
Department of Histopathology, Cork University Hospital, Wilton, Cork, Eire, Ireland. jansmichael@gmail.comIssue Date
2010-01MeSH
AdultAorta, Thoracic
Aortitis
Cogan Syndrome
Female
Giant Cell Arteritis
Humans
Hypertension, Pulmonary
Middle Aged
Vasculitis
Wegener Granulomatosis
Metadata
Show full item recordCitation
Thoracic vasculitis presenting as surgical problems. 2010, 456 (1):91-6 Virchows Arch.Journal
Virchows Archiv : an international journal of pathologyDOI
10.1007/s00428-009-0865-0PubMed ID
20012090Abstract
We present four patients with vasculitis manifesting with unusual clinical or pathological features, generating surgical problems. Two cases presented with pulmonary hypertension, with investigations and radiological evidence prompting clinical suspicion of pulmonary thrombo-embolic disease. First case, with an antecedant history of Wegener's granulomatosis (WG), demonstrated following "embolectomy", WG involving the large pulmonary elastic arteries. The second case of inoperable "pulmonary thrombo-embolic disease" was subsequently found at limited post mortem to have giant cell arteritis, which affected widespread small peripheral pulmonary arterial vessels. The other two cases were of aortitis occurring in the background of immune-mediated disease, which had been treated with aggressive immunosuppression regimens. The first of these was a case of Cogan's syndrome complicated by descending aortitis, a rarely reported phenomenon, with co-existent acute endocarditis of the aortic valve leaflets. Most cases of endocarditis in this context occur secondary to and in continuity with ascending aortitis. That this case, and a case of ascending aortitis occurring in the context of relapsing polychondritis occurred in the face of aggressive immunosuppression with an apparent clinical response, underscores the need to not accept a clinical picture at face value. This has implications for clinical management, particularly in the follow-up of surgical prosthetic devices such as grafts which may be used in these cases. All four cases emphasise the continued importance of histology and the post-mortem examination in elucidating previously undetected or unsuspected disease.Item Type
ArticleLanguage
enISSN
1432-2307ae974a485f413a2113503eed53cd6c53
10.1007/s00428-009-0865-0
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